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Vol. 06, No. 02, pp. 342 –357 (2025) ISSN: 2708-0757 JOURNAL OF APPLIED SCIENCE AND TECHNOLOGY TRENDS www.jastt.org 342 doi: 10.38094/jastt62404 A Hybrid LLM–Knowledge Graph Framework for Accurate Biomedical Question Answering Havraz Y. Omar1,²*, Abdulhakeem O. Mohammed³ ¹Department of Information Technology, Technical College of Duhok, Duhok Polytechnic University, Duhok, Kurdistan Region, Iraq. havraz.omar@dpu.edu.krd ² Department of Information Technology, Technical College of Informatics – Akre, Akre University for Applied Sciences, Akre, Kurdistan Region, Iraq. ³ Department of Computer Science, College of Science, University of Zakho, Zakho, Kurdistan Region, Iraq. a.mohammed@uoz.edu.krd *Correspondence: havraz.omar@dpu.edu.krd Abstract Biomedical question answering requires accurate and interpretable systems; however, existing approaches often face challenges such as language model hallucinations and limited reasoning when relying solely on standalone knowledge graphs. To address these limitations, this study proposes a hybrid framework that integrates the LLaMA-3B language model with a Neo4j-based drug–disease–symptom knowledge graph. The system translates natural language questions into executable Cypher queries, operates on an iBKH-derived graph comprising over 65,000 entities and 3 million relationships, and returns answers with supporting evidence through a transparent interface. Experiments conducted on 60 biomedical questions across three levels of difficulty demonstrate the robustness of the approach: 96% exact match for simple queries, 95% for medium queries, and 86.7% for complex queries. Overall, the system achieves Precision@5 of 96.1%, Recall@5 of 89.0%, F1@5 of 91.0%, Hits@k of 96.1%, and an MRR of 94.4%, while maintaining an average response time of only 6.07 seconds. These results indicate that the system retrieves nearly all relevant answers, ranks them correctly, and delivers them with latency low enough for interactive use. Moreover, unlike cloud-based APIs such as ChatGPT, which require internet connectivity and external data transmission, the proposed framework operates fully offline, ensuring privacy, reproducibility, and compliance with biomedical data governance. Overall, this pipeline provides an accurate, efficient, and privacy-preserving solution for biomedical question answering, making it a practical alternative to cloud-dependent approaches in sensitive healthcare contexts. Keywords: Knowledge Graph, LLM, Question Answering, Neo4j, Biomedical Informatics, Healthcare AI, LLaMA 3. Received: August 14th, 2025 / Revised: October 10th, 2025 / Accepted: October 16th, 2025 / Online: October 20th, 2025 I. INTRODUCTION Answering questions in the biomedical field is a difficult task due to the complexity of medical knowledge and the need for precision. In recent years, large language models (LLMs) like LLaMA, GPT-4 have made progress in understanding and generating human-like responses to medical questions [1, 2]. These models can process large amounts of information and respond in natural language, which makes them helpful in healthcare settings [3]. However, they often struggle to provide accurate answers when dealing with specialized biomedical content [4, 5]. One major issue with LLMs is a problem called hallucination, where the model generates information that sounds right but is actually incorrect or unsupported [6]. In medical applications, this can be dangerous, as healthcare professionals rely on precise and trustworthy information [7]. Therefore, researchers are exploring ways to combine LLMs with structured sources of knowledge to improve their reliability [8]. LLM-only systems in biomedicine still hallucinate and are hard to verify, limiting safe use [9, 10]. Biomedical knowledge graphs (BKGs) such as iBKH and SPOKE curate multi-source facts and enable multi-hop reasoning, yet they neither interpret free text nor generate answers [11, 12]. Recent hybrids (KG- aware RAG) improve grounding but often lack explicit path-level justifications and robust end-to-end answer evaluation [13, 14]. Recent studies have increasingly integrated Knowledge Graphs (KGs) with Large Language Models (LLMs) to improve factual accuracy, reasoning, and reduce hallucinations. Notable examples include DR.KNOWS, which combines UMLS-based KGs with LLMs for better diagnostic reasoning [15], KnowNet
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Omar & Mohammed / Journal of Applied Science and Technology Trends Vol. 06, No. 02, pp. 342 –357 (2025) 343 for visualizing and validating LLM outputs [16], and MedKA for KG-enhanced question answering [17]. To address these challenges, several recent works have explored the integration of large language models with biomedical knowledge graphs (KGs). A biomedical KG is a structured network that connects entities such as diseases, drugs, and symptoms using defined relationships [18, 19]. These graphs store verified medical knowledge from trusted databases, allowing for more accurate and explainable responses [12]. KGs are especially useful in multi-step reasoning tasks, where finding an answer requires connecting different pieces of information [20]. These entities and relationships can be visually represented in a biomedical knowledge graph, as shown in Fig. 1, where nodes represent medical concepts such as drugs, diseases, symptoms, and pathways, and edges denote their semantic relationships. Fig. 1. Overview of Biomedical Knowledge Graph Entities and Relationships One example of a widely used biomedical KG is SPOKE, which includes millions of nodes and relationships from over 40 biomedical databases [12]. Integrating an LLM with a KG allows the strengths of both technologies to work together: the LLM provides language understanding, and the KG provides structured, factual knowledge [21, 22]. A common method is retrieval-augmented generation (RAG), where the LLM retrieves information from the KG and uses it to generate more accurate responses [13, 23]. In more advanced setups, the LLM can even generate queries like Cypher to fetch specific data from the graph [24, 25]. Neo4j is a popular graph database that supports fast and flexible storage and querying of knowledge graphs using Cypher [21]. It is well-suited for biomedical applications because it allows easy exploration of complex medical relationships. Recent work has shown that combining Neo4j with LLMs can lead to better accuracy, fewer hallucinations, and more explainable results [24, 26]. Despite improvements, building a reliable hybrid system that combines an LLM with a biomedical KG remains a technical challenge. Some approaches require complex pipelines or large training datasets, while others rely on fine-tuning specific to a narrow set of questions [27, 28]. There is still a need for systems that are both accurate and easy to scale, especially in domains like medicine where the cost of errors is high [22]. Recent advances in KG-augmented LLMs have improved performance, yet biomedical QA continues to face three practical gaps: 1. Traceability: LLM-only or text-retrieval-only pipelines rarely provide graph-grounded justifications; users lack the ability to inspect the exact nodes and edges that support an answer. 2. Evaluation: Prior work often judges quality via surface- form checks (e.g., matching a Cypher template), which fails to capture end-to-end answer correctness or ranking quality across different difficulty levels. 3. Deployment: Many solutions assume cloud resources or domain-specific fine-tuning, yet biomedical contexts typically demand a local, privacy-preserving system with low latency and reproducible behavior. Timestamp-aware execution and periodic KG refresh help avoid deprecated or contraindicated links, making the tool better suited for safety-critical clinical contexts (e.g., drug–drug interactions). To address these limitations, Our work introduces a locally deployable pipeline that translates biomedical questions into executable Cypher queries over a Neo4j knowledge graph. The system returns answers with supporting nodes and edges, and is
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Omar & Mohammed / Journal of Applied Science and Technology Trends Vol. 06, No. 02, pp. 342 –357 (2025) 344 evaluated using Exact Match, Precision, Recall, F1, Hits@k, MRR, and latency across simple, medium, and complex question sets. Unlike prior template-based methods, our approach enables traceable, outcome-level validation. In summary, the main contributions of this work are as follows: • Hybrid LLM to Cypher QA: A system that translates natural language questions into accurate, executable Cypher over a Neo4j drug, disease, and symptom KG. • Prompt-driven query generation: Schema, entity aware prompting that reliably maps diverse biomedical questions to graph queries. • Evidence transparency: Along with each answer, we surface the generated Cypher and the supporting justification subgraph (nodes, edges) plus a brief natural language rationale. • Answer-level evaluation: End-to-end assessment using Exact Match, F1, Precision/Recall, Hits@k, MRR and latency across simple, medium and complex tiers. • Local, reproducible deployment: On-prem LLaMA 3 inference (no cloud dependency) suitable for biomedical settings requiring low latency and strong data control. The remainder of this paper is organized as follows: Section 2 reviews related work on biomedical knowledge graphs and LLM-based QA systems. Section 3 provides background on knowledge graphs, large language models, and question answering frameworks. Section 4 details the proposed methodology, including system architecture, dataset construction, and query translation. Section 5 presents the experimental results through both quantitative metrics and qualitative case studies. Section 6 discusses the findings, analyzes limitations, and compares performance against baseline models. Finally, Section 7 concludes this paper and outlines directions for future work. II. RELATED WORK Recently, studies have concentrated on the integration of clinical and medical knowledge graphs (LLM) to improve the answer to medical questions. Researchers have derived several biomedical KGs using Neo4j and incorporated the application of LLMs like LLaMA and GPT to convert natural language questions into graph queries. Improvements in answer correctness, reduction of hallucination errors, one-to-many relationships, and support for complex reasoning were the objectives of these efforts. Some frameworks also adopted retrieval methods to ground responses in secure data. Su et al.[11] developed an integrative Biomedical Knowledge Hub (iBKH), a huge biomedical knowledge graph that comprises 18 of the very best data sources. The deployment of the iBKH in Neo4j allows for a user-friendly web portal to allow fast and interactive knowledge retrieval. The system implemented advanced graph learning techniques to enable the discovery of biomedical knowledge, illustrated by an example of repurposing in silico drugs for Alzheimer’s disease. iBKH achieved promising predictive performance for known drugs and proposed possible new drug candidates. Rajabi and Kafaie[19] proposed a disease knowledge graph using a cross-referential disease database comprising diseases, symptoms, and drugs interconnected with relationships. They transferred the data into Neo4j to create a graph of 9,400 nodes and 45,000 relationships representing the semantic links between medical concepts. Applying Cypher queries enabled answering complex medical questions regarding identifying drugs that may cause certain diseases; it was demonstrated that the graph inferred new relationships not explicitly existing in the original data. The conclusion was that disease knowledge graphs sped up clinical discovery and contributed to understanding complex medical relationships. Hou et al.[3] assessed and contrasted ChatGPT (both GPT- 3.5 and GPT-4) and the biomedical knowledge graphs (BKGs) concerning their ability to answer biomedical questions, generate new knowledge, and reason. Their datasets were focused on dietary supplements and drugs, while evaluation criteria entailed accuracy, novelty, and reasoning ability. The results indicate that while GPT-4 surpassed GPT-3.5 and BKGs in knowledge provision, it proved inconsistent with regard to citations and reasoning. Compared to them, BKGs scored higher in accuracy and reliability, especially in discovering novel links as well as within structured reasoning. Soman et al.[13] presented a novel framework called KG- RAG that integrates a large biomedical knowledge graph (SPOKE) with LLaMA 2, GPT-3.5, and GPT-4 (LLMs) to produce accurate biomedical text. They optimized the retrieval of relevant graph context to cut over 50% tokens without losing accuracy. It aided LLMs in performing better on biomedical question answering with very high accuracy boosts, especially in the case of LLaMA 2. They compared KG-RAG to other retrieval methods and indicated its comparatively more robust and efficient results. The framework produced reliable evidence- based answers grounded in biomedical knowledge. Luo et al.[23] created ChatKBQA, a new framework with a question-and-answer approach over knowledge bases that first generates logical forms with the help of fine-tuned LLMs and then retrieves the relevant entities and relations. This generate- then-retrieve approach is supposed to handle a couple of issues with the earlier methods concerning tedious retrieval and error propagation. They fine-tuned open-source LLMs like LLaMA 2 to change natural-language questions into logical forms with high accuracy. The retrieval phase uses unsupervised phrase-level semantic matching in a way that enhances the alignment of entities and relations. Experiments on benchmark datasets indicate ChatKBQA to be superior to its predecessors, with the highest accuracy to date. Pusch and Conrad[6] conducted work under a hybrid approach conflating LLMs and biomedical Knowledge Graphs (KGs) to suppress hallucination errors in question-answering. They proposed query-checking algorithms for validating, correcting, and executing the KG Cypher queries that LLMs generated, thereby attaining accurate and understandable answers. The system used retrieval-augmented generation (RAG) to ground answers within KG data. The methodology was validated on a biomedical KG called PrimeKG using 50 benchmark questions, assessing models like GPT-4 Turbo and LLaMA 3. Commercially available GPT-4 Turbo obtained
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Omar & Mohammed / Journal of Applied Science and Technology Trends Vol. 06, No. 02, pp. 342 –357 (2025) 345 record-high accuracy, while open-source models achieved impressive gains through prompt optimization. Feng et al.[22] developed the Knowledge Graph-based Thought (KGT) framework that integrated LLMs with a pan- cancer knowledge graph for biomedical question answering. KGT was designed to reason on the knowledge graph schema and identify optimal subgraphs to use for directing accurate answer generation, all without fine-tuning the LLMs. The framework is benchmarked against a new dataset (PcQA) designed specifically for pan-cancer KGQA tasks and has outperformed all existing state-of-the-art approaches by a rather large margin. KGT’s practicality in biomedical issues was highlighted through case studies for drug repositioning, drug resistance, and biomarker discovery. Their approach exhibited robust adaptability among various LLMs. Rezaei et al.[26] developed AMG-RAG, a dynamic framework that utilizes autonomous LLM agents with medical search tools in the continuous construction and real-time updating of Medical Knowledge Graphs (MKGs). Their system incorporated confidence scoring and multi-hop reasoning to improve accuracy and interpretability in medical question answering. AMG-RAG outperformed size models on both very hard MEDQA benchmarks and more accessible MedMCQA ones, proving that it could conduct efficient reasoning based on current structured medical knowledge. They also used Neo4j to manage the knowledge graphs while adding external searches to ensure the latest data. Tiwari et al.[24] presented Auto-Cypher, a recent automated pipeline for producing high-quality synthetic data for training LLMs by mapping natural language to Cypher queries for graph databases like Neo4j. The pipeline deployed the novelty of LLM- as-database-filler to synthesize Neo4j databases for the execution of generated queries to ensure their correctness. A sizable dataset called SynthCypher was created, spanning multiple domains and complex queries, leading to a 40% improvement in LLM performance on Cypher generation. The datasets were used to fine-tune open-source models such as LLaMA, Mistral, and Qwen, and the SPIDER benchmark was adapted for evaluation purposes. Mohammed et al.[29] proposed a hybridized GraphRAG framework combining Neo4j-based UMLS knowledge graphs with a vector store for medical textbooks to create an improved U.S.M.L.E.-style clinical question-answering approach. The project integrated symbolic reasoning from knowledge graphs with semantic retrieval performed on text embeddings to enhance relevance and accuracy via adaptive re-ranking and query expansion. The system had the answers produced by GPT-4o- Mini, with different prompting strategies encouraging evidence- based and traceable responses grounded in verified medical knowledge. Experiments showed that the hybrid approach improved factual accuracy and citation fidelity as compared to the L.L.M.-only approach, enhancing transparency and reliability. It is shown that binding both structured and unstructured medical knowledge sources could aid in ameliorating hallucinations and hence improve clinical trustworthiness in AI-driven medical QA. Yang et al.[30] articulated sepsis knowledge graph was crafted by combining multicenter clinical data from over 10,000 patients with the help of GPT-4 for entity recognition and relationship extraction. Real-world data were collected from three hospitals and integrated with clinical guidelines and databases from the public domain. The knowledge graph contained 1,894 nodes and 2,021 relationships pertaining to diseases, symptoms, biomarkers, treatments, and complications. GPT outperformed other models in every resolution on sepsis- specific datasets to obtain high F1-score results. The constructed graph highlighted complex interactions in sepsis for assisting clinical decision-making and was implemented on Neo4j. Guan et al.[20] proposed a novel method for constructing a local knowledge graph from retrieved biomedical documents by extracting propositional claims. They carried out layer wise summarization on this graph to capture multi-document relationships and provide comprehensive contextual information to a language model for question-answering purposes. The method resolved issues in multi-document biomedical QA, such as noise cancellation and efficient context usage. They then tested their method on several benchmarks for biomedical question answering, achieving performance at least comparable to, if not better than, existing retrieval-augmented generation (RAG) baselines. The study established enhanced reasoning and answer accuracy of the model achieved through structured graph summarization. Previous studies have improved biomedical QA using KGs and LLMs, but important gaps remain. Most systems lack transparent, graph-based justifications, rely on limited evaluation methods, or depend on cloud resources that reduce privacy and reproducibility. Our framework addresses these gaps by providing visible Cypher queries with evidence subgraphs, applying comprehensive performance metrics across difficulty levels, and ensuring fully local, privacy-preserving deployment. Table I summarizes key previous studies on biomedical knowledge graphs and question answering, outlining their methods, datasets, and main limitations. III. PRELIMINARIES This section outlines the fundamental concepts required to understand the proposed system. It introduces biomedical knowledge graphs, explains how Neo4j stores data in graph form, and describes the use of Cypher for querying. It also provides a brief overview of large language models (LLMs) and their role in interpreting natural language. A. Biomedical Knowledge Graphs Biomedical Knowledge Graphs (BKGs) provide a structured representation of complex biomedical information by modeling diverse medical entities, such as diseases, drugs, symptoms, and biological pathways, as interconnected nodes within a graph structure. The edges in these graphs represent the semantic relationships between these entities, including ’treats’, ’causes’, ’interacts with’ and many others, as illustrated in Fig 1. This form of representation enables the integration of heterogeneous biomedical data from a wide range of sources, including
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Omar & Mohammed / Journal of Applied Science and Technology Trends Vol. 06, No. 02, pp. 342 –357 (2025) 346 scientific literature, clinical records, genomic databases, and experimental findings [19, 31]. Such integration creates a comprehensive biomedical knowledge base that supports advanced analytics and discovery. For example, biomedical knowledge graphs can reveal previously unknown relationships (e.g., between drugs and diseases) and help prioritize potential biomarkers for complex conditions. The Integrative Biomedical Knowledge Hub (iBKH) is one such large-scale graph that consolidates diverse biomedical resources into a unified hub, enabling discovery at scale [11]. Beyond iBKH, large biomedical knowledge graphs such as SPOKE further illustrate how graph integration accelerates research and supports precision-medicine use cases [12]. Overall, these graphs serve as foundational resources for data-driven and personalized medicine. These knowledge graphs serve as foundational resources for precision medicine, where treatment can be tailored to the individual’s biological profile, improving outcomes and minimizing side effects [19, 31]. TABLE I. SUMMARY OF RELATED RESEARCH ON BIOMEDICAL KGS AND QUESTION ANSWERING Ref. Year Data/Graph Method Baselines Key Metric Limitation [11] 2023 iBKH (18 biomedical sources, Neo4j) Integrative KG + Graph learning; drug repurposing case Known drugs, Alzheimer’s study Predictive performance (drug repurposing) Limited to Alzheimer’s case study; scalability and updates not detailed [19] 2023 Disease KG (9,400 nodes, 45,000 relations in Neo4j) Cypher queries for disease– drug–symptom reasoning Cross-referential disease DB New relation inference; complex query answering Limited to single domain; lacks large-scale evaluation [3] 2023 BKGs vs. GPT-3.5/4 Comparative QA study: LLMs vs. KGs GPT-3.5, GPT-4, KG reasoning Accuracy, Novelty, Reasoning GPT-4 inconsistent in reasoning/citations; KG less fluent but more reliable [13] 2024 SPOKE KG + LLaMA2, GPT-3.5, GPT-4 KG-optimized retrieval for LLMs (RAG) Other retrieval methods Accuracy, token reduction >50% Focus on retrieval optimization, not KG construction [23] 2024 Benchmark KB datasets Generate-then-retrieve (LLM → logical form → KB retrieval) Prior KBQA methods Accuracy (highest to date) Risk of error in logical form generation [6] 2024 PrimeKG LLM + KG hybrid, Cypher query validation, RAG GPT-4 Turbo, LLaMA 3 Accuracy, Explainability Dependent on KG coverage; computationally intensive [22] 2025 Pan-cancer KG (PcQA dataset) KG-enhanced reasoning (subgraph selection) SOTA KGQA methods Outperformed SOTA on PcQA Limited to pan-cancer focus; no fine-tuning explored [26] 2025 Dynamic Medical KG + Neo4j LLM agents + multi-hop reasoning MEDQA, MedMCQA baselines Accuracy, Interpretability High system complexity; requires continuous updating [24] 2025 SynthCypher dataset (Neo4j + synthetic queries) LLM-supervised Cypher generation and verification SPIDER benchmark Cypher accuracy 40% Synthetic dataset may not capture all real-world cases [29] 2025 UMLS KG + Neo4j Hybrid GraphRAG LLM-only QA Accuracy, Citation fidelity More complex pipeline; relies on external vector store [30] 2025 Clinical data (10k patients, 1,894 nodes, Neo4j) KG construction using GPT-4 for entity/relation extraction Other KG construction methods High F1-scores Focus only on sepsis; limited generalization [20] 2025 Local KG from biomedical documents Multi-level summarization over KG for QA RAG baselines QA accuracy, reasoning Tested mainly on document QA; scalability not proven B. Neo4j Graph Database To manage the complexity and large size of biomedical knowledge graphs, specialized graph databases are needed. Neo4j is one of the most popular graph databases designed to store and query data structured as nodes (entities) and relationships (edges), both of which can have descriptive properties [32, 33]. It uses the property graph model, which makes it easy to represent complex, connected biomedical data such as drug-gene interactions or disease pathways. Neo4j’s Cypher query language is especially advantageous because it allows users to write expressive and efficient queries to explore multi-step connections in the data [34]. Neo4j works well for biomedical data because it can quickly run complicated queries over highly interconnected datasets. This is important in biology and medicine, where relationships between entities like proteins, diseases, and drugs are often complex and layered. Studies have shown that Neo4j handles large biomedical graphs efficiently, making it a favorite among researchers and industry users alike [33, 35, 36]. Its indexing and caching mechanisms also help speed up query processing and data retrieval [37]. Moreover, Neo4j integrates smoothly with many programming languages and analytics tools, which makes it easier to build interactive biomedical applications and clinical decision support systems that can turn complex graph data into useful insights [38, 39]. C. Large Language Models (LLMs) in Biomedical Question Answering Large Language Models (LLMs) are powerful AI systems trained on vast amounts of text data. They learn the structure and patterns of language, enabling them to understand questions, generate responses, summarize information, and perform other complex language tasks. Well-known models such as LLaMA and GPT-3 have greatly advanced the field of natural language processing by showing strong performance across many tasks [40, 41].
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Omar & Mohammed / Journal of Applied Science and Technology Trends Vol. 06, No. 02, pp. 342 –357 (2025) 347 In biomedical research and clinical settings, LLMs help translate natural language questions from doctors, researchers, or patients into precise, structured queries that can be executed on biomedical knowledge graphs and databases. This makes it easier to retrieve detailed biomedical information like drug interactions, gene-disease associations, and symptom descriptions [42, 43]. Despite their power, LLMs can sometimes generate incorrect or fabricated responses, a phenomenon known as hallucination, which poses risks in sensitive biomedical contexts. These hallucinations occur because the models generate plausible answers based on patterns learned from data rather than verified knowledge. To mitigate this, researchers integrate LLMs with biomedical knowledge graphs to ground answers in factual data, significantly improving accuracy and reducing misinformation [4]. Further improvements come from fine-tuning LLMs on biomedical corpora and carefully engineering prompts, which enhance their reliability and relevance in medical question answering. Additionally, combining LLMs with knowledge graphs and reasoning techniques is an active area of research that promises to increase the interpretability and trustworthiness of AI systems in biomedicine. These advances are critical for developing tools that assist clinical decision-making and accelerate biomedical discoveries [43, 44]. IV. METHODS AND MATERIALS This section describes the methodology used to build a biomedical question-answer system. The proposed method consists of three main stages; First, a biomedical knowledge graph is constructed in the data ingestion phase, using structured data sources (e.g., diseases, drugs, symptoms). Second, a language model (LLaMA 3) interprets the user’s question written in English in the user interaction phase and converts it into a Cypher query. Third, a graphical user interface allows users to type questions and view the results interactively. A. System Architecture The proposed framework is organized as a step-by-step pipeline that integrates a local large language model (LLM) with a biomedical knowledge graph stored in Neo4j. The overall workflow is illustrated in Fig. 2. Each module performs a specific function, and together they ensure that the system delivers accurate, reliable, and explainable answers. Fig. 2. Workflow of the LLaMA 3 and Neo4j-Based QA System Step 1. User Input (Flask Web Interface): A user submits a biomedical question in natural language through a Flask-based web interface. The same interface will later display the answer, the executed query, and a compact preview of the retrieved rows. Step 2. LLM Processing and Initial Cypher: The text query is forwarded to a local LLaMA 3 module, which interprets the intent and drafts an initial Cypher pattern suitable for querying the graph. Step 3. Cypher Query Generation: The Cypher Query Gen block receives the initial pattern from LLaMA 3, canonicalizes and completes it (projection, DISTINCT, filters), and returns a finalized Cypher query to the model. Step 4. Query execution on Neo4j: LLaMA 3 passes the finalized query to the Query execution component (inside the ’Answer modules’ box), which runs it against the Neo4j Knowledge Graph. KG supplies knowledge graph data (e.g.,
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Omar & Mohammed / Journal of Applied Science and Technology Trends Vol. 06, No. 02, pp. 342 –357 (2025) 348 drugs, diseases, symptoms) and execution returns structured data (tuples) relevant to the question. Step 5. Answer Synthesis: The structured tuples flow to Answer Synthesis, which aggregates and formats them into a concise raw answer. This raw answer is sent back to LLaMA 3 to optionally refine the phrasing while preserving the retrieved facts. Step 6. Result Presentation: LLaMA 3 produces the final answer, which the interface displays together with the executed Cypher query and an optional preview of the returned rows, improving transparency and trust. The pipeline couples LLM-based language understanding (LLaMA 3) with a schema-grounded Neo4j knowledge graph. The Cypher Query Gen refines the query formulation, Query Execution retrieves evidence and Answer Synthesis converts structured results into readable outputs that produce answers that are accurate, interpretable, and easy to audit directly from the displayed query and evidence. B. Dataset and Knowledge Graph Construction 1) Dataset The integrated Biomedical Knowledge Hub (iBKH), a large biomedical knowledge base, forms the first level of the system and integrates information from various curated high-quality biomedical databases. This implies that the data set includes various types of entities, such as diseases, symptoms, drugs, biological pathways, etc. This study used the representative subset of the iBKH dataset, which contained 65828 biomedical entities. These entities are semantically interconnected through a total of 3004166 relationships, thus creating a rich knowledge graph. The iBKH dataset was originally introduced in [11], and it is freely available at (https://github.com/wcm-wanglab/iBKH). This dataset is the core semantic foundation upon which this study is built. The knowledge graph is populated from multiple tabular sources (CSV files), each listing entities or relationships. The main input files and their contents are as follows: • Disease vocabulary(disease_vocab.csv): Contains columns such as primary (a unique disease ID), name, do_id (Disease Ontology ID), kegg_id, and umls_cui (UMLS Concept ID). Each row represents a disease node with external identifiers. • Drug vocabulary (drug_vocab.csv): Includes primary (unique drug ID), name, drugbank_id, kegg_id, pharmgkb_id, umls_cui, mesh_id, iDISK_id and CID (PubChem ID). Each row defines a drug node with standard database identifiers. • Symptom vocabulary (symptom_vocab.csv): Contains primary (unique symptom ID), name, mesh_id, umls_cui and iDISK_id. Each row defines a symptom node. • Side effect vocabulary (side_effect_vocab.csv): Includes primary (unique side-effect ID) and name. Each row represents a side-effect node (with UMLS ID when available). • Pathway vocabulary (pathway_vocab.csv): Contains primary (unique pathway ID), name, reactome_id, go_id, and kegg_id. Each row defines a biological pathway node. Relationship files (each row typically contains two entity IDs and one or more boolean flags or codes) include: • Disease–Symptom links (Di_Sy_res.csv): Rows include Disease and Symptom IDs, a presence flag (1 or 0) and a data source. If Present = 1, a HAS_SYMPTOM edge is created from the disease to the symptom, with properties for presence and source. • Disease–Disease links (di_di_res.csv): Rows include Disease_1 and Disease_2 IDs with binary flags for is_a and Resemble. If is_a = 1, an (IS_A) edge is created (Disease_1 → Disease_2); if Resemble = 1, a RESEMBLES edge is created. The source field is used for provenance. • Drug–Disease links (D_Di_res.csv): Includes Drug and Disease IDs with several binary flags. If a flag equals 1, a corresponding edge is created: o TREATS (Treats = 1) o PALLIATES (Palliates = 1) o ASSOCIATED_WITH (Associate = 1) o ALLEVIATES_REDUCES (alleviates = 1) o TREATMENT_THERAPY (treatment/therapy = 1) o INHIBITS_CELL_GROWTH (inhibits cell growth = 1) o HAS_BIOMARKER (biomarkers = 1) o PREVENTS_SUPPRESSES (prevents/suppresses = 1) o ROLE_IN_PATHOGENESIS (role in disease pathogenesis = 1) • Drug–SideEffect links (D_SE_res.csv): Contains Drug and SideEffect IDs with a Source column. Each row creates a CAUSES edge from the drug to the side effect, with source as an edge property. • Drug–Drug interactions (D_D_res.csv): Rows include Drug_1 and Drug_2 IDs with flags for Interaction and Resemble. If Interaction = 1, an INTERACTS_WITH edge is created (bidirectional). If Resemble = 1, a RESEMBLES edge is added. • Drug–Pathway links (D_Pwy_res.csv): Includes Drug ID and Pathway ID. Each row generates an ASSOCIATED_WITH edge from the drug to the pathway. • Disease–Pathway links (Di_Pwy_res.csv): Contains Disease ID and Pathway ID. Each row creates an ASSOCIATED_WITH edge from the disease to the pathway. 2) Data Upload Performance The time required to upload different types of entities and relationships into the Neo4j biomedical knowledge graph, measured in seconds. These measurements reflect both the size and complexity of the data being processed. As shown in Table II, the longest upload time is for Drug- Drug Relationships, which takes approximately 190 seconds due to the large number of edges (over 3 million). Following this, Disease-Disease and Drug-Disease Relationships also require considerable time for loading. On the other hand, individual
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Omar & Mohammed / Journal of Applied Science and Technology Trends Vol. 06, No. 02, pp. 342 –357 (2025) 349 entities such as Diseases and Drugs are uploaded much faster, generally under 2 seconds. TABLE II. DATA UPLOAD TIMES FOR DIFFERENT ENTITY AND RELATIONSHIP TYPES IN NEO4J Entity / Relationship Type Upload Time (seconds) Disease 0.81 Drugs 1.08 Symptoms 0.06 Side Effects 0.14 Pathways 0.08 Disease-Disease Relationships 30.97 Drug-Disease Relationships 30.28 Drug-SideEffect Relationships 5.24 Drug-Drug Relationships 190.09 Drug-Pathway Relationships 0.14 Disease-Pathway Relationships 0.06 Disease-Symptom Relationships 0.12 Fig. 3, presents a vertical bar chart that visually compares these upload times across the different entity and relationship types. The chart clearly shows the significant difference in upload duration between nodes and edges, emphasizing the higher cost of ingesting complex relationships in the graph. 3) Experimental Environment In this study, the proposed biomedical question answering system was evaluated using a locally hosted environment. All experiments were conducted on a Windows 11 Pro (64-bit) system equipped with an Intel Core i5-10500H processor running at 2.50 GHz (12 logical CPUs), 24 GB of RAM, and an NVIDIA GeForce GTX 1650 GPU with Max-Q Design. The Neo4j graph database (v4.4.5) was managed through Neo4j Desktop (v1.6.2), and the LLaMA 3B language model was executed locally using optimized configurations suitable for this hardware setup. Each Cypher query generated by the system was executed multiple times to calculate an average response time, ensuring consistency across varying levels of question difficulty. The knowledge graph was constructed using the iBKH dataset, and data loading and system performance were carefully monitored to maintain stability during testing. This experimental setup provides a reliable and reproducible environment for benchmarking the effectiveness and responsiveness of the hybrid QA system. Fig. 3. Upload times for various biomedical entities and relationships in Neo4j. We run LLaMA 3 locally (not via cloud APIs) to satisfy biomedical privacy/governance (no data leaves the host) and to maximize reproducibility (fixed GGUF checkpoint, pinned llama.cpp commit, controlled seeds settings, constant hardware). Local execution yields predictable cost availability and stable latency (no network jitter) and lets us enforce executable Cypher grounding with per edge provenance and timestamp aware execution. 4) Knowledge Graph(KG) Construction The Neo4j graph database was used as the backend to store and query the KG, and it is a graph database designed for highly connected data. Before loading data, uniqueness constraints were created on the primary property for each node label (Disease, Drug, Symptom, SideE ffect, Pathway). This enforces that each primary ID appears only once, preventing duplicate entities. For efficient lookups in queries, a search index was created based on the name property of each node label. As noted in the Neo4j
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Omar & Mohammed / Journal of Applied Science and Technology Trends Vol. 06, No. 02, pp. 342 –357 (2025) 350 documentation, indexes “enable quicker and more efficient pattern matching” by allowing the query planner to rapidly locate nodes by label and property. With the schema in place, data was imported using Cypher’s LOAD CSV commands. For each vocabulary file, rows with nonnull primary IDs were processed: the code used MERGE to create (or match existing) nodes with the given label and primary property, and then SET the remaining attributes from the CSV columns. For example, in importing disease_vocab.csv, each row produced a node (:Disease primary: <id>) with properties name, do_id, kegg_id, and umls_cui set from the row (empty strings were turned into null). Similarly, drug_vocab.csv rows produced drug nodes with properties for DrugBank, KEGG, PharmGKB IDs, etc. This approach follows best practice: using MERGE on the unique ID ensures that nodes are not duplicated during multiple passes. After all nodes were created, the relationship CSVs were loaded. Each row in those files was matched to the corresponding source and target nodes by their primary IDs, and conditional logic was used to create edges. For example, the disease- symptom file (Di_Sy_res.csv) was processed by matching a disease node and a symptom node for each row, then executing MERGE (d)-[r:HAS_SYMPTOM]-(s) if the present column is nonzero; the edge was given a present property and a source property from the CSV. The disease-disease file (di_di_res.csv) was processed by matching disease nodes d1 and d2:If is_a = 1, a (:Disease)-[:IS_A]->(:Disease) edge was merged; if Resemble = 1, a (:Disease)-[:RESEMBLES]->(:Disease) edge was merged. Drug-disease relationships were handled similarly: the script matched a Drug node and a Disease node for each row of D_Di_res.csv, then for each flag that was 1, it merged the corresponding edge label (such as TREATS, PALLIATES, ASSOCIATED_WITH, etc.) from the drug to the disease. Drug– side-effect rows produced (:Drug)-[:CAUSES]->(:SideEffect) edges with the source noted, and drug–drug rows produced either INTERACTS_WITH or RESEMBLES edges between matched drug pairs. Finally, the drug-pathway and disease-pathway files each produced:ASSOCIATED_WITH edges linking drugs or diseases to pathway nodes. In this graph model, most relationships are directional (for example, a drug TREATS a disease; a disease does not TREAT a drug). This follows common practice in biomedical KGs. The same relations (such as RESEMBLES or INTERACTS_WITH) are inherently symmetric, but were stored as two directed edges or one undirected edge depending on implementation. All relationship creation steps used Cypher’s MERGE so that repeated loads or out-of-order imports did not create duplicate edges. This study used a static KG snapshot for reproducibility, but the system also supports incremental updates through the Neo4j MERGE and batch import functions. A summary of the different types of nodes and relationships is provided, together with their counts, descriptions, and examples in Table III. TABLE III. DETAILED SUMMARY OF KNOWLEDGE GRAPH COMPONENTS IN IBKH SUBSET Component Type Entity/Relationship Count Description Examples Disease 19,236 Medical conditions and disorders Diabetes, Hypertension, Cancer Drug 37,997 Pharmaceutical compounds Aspirin, Insulin, Amoxicillin Nodes Symptom 1,361 Clinical signs and symptoms Headache, Fever, Nausea SideEffect 4,251 Negative outcomes of drugs Nausea, Drowsiness Pathway 2,983 Biological processes and pathways Apoptosis, Glycolysis ASSOCIATED_WITH 101,534 General associations (disease– pathway, drug–pathway, etc.) Diabetes ASSOCIATED_WITH Pathway CAUSES 145,321 Drug-SideEffect relationships Aspirin CAUSES Gastric_Bleeding HAS_SYMPTOM 3,357 Disease-symptom links COVID-19 HAS_SYMPTOM Fever HAS_BIOMARKER 412 Biomarkers linked to disease PSA HAS_BIOMARKER Prostate_Cancer Relationships INHIBITS_CELL_GROWTH 1,508 Drugs inhibiting cell growth Chemo INHIBITS_CELL_GROWTH Tumor INTERACTS_WITH 2,682,142 Drug-drug interactions Aspirin INTERACTS_WITH Warfarin IS_A 10,529 Subtype hierarchy Flu IS_A Viral_Infection PALLIATES 388 Drug palliates disease Morphine PALLIATES Cancer PREVENTS_SUPPRESSES 859 Preventive links Vaccine PREVENTS_SUPPRESSES Measles RESEMBLES 7,000 Similarity relationships DrugA RESEMBLES DrugB TREATMENT_THERAPY 44,852 Therapy relationships Radiotherapy TREATMENT_THERAPY Tumor TREATS 5,491 Drug-disease links Insulin TREATS Diabetes ALLEVIATES_REDUCES ∼180,000 Symptom relief Paracetamol ALLEVIATES_REDUCES Fever Total Nodes 65,828 Total biomedical entities — Relationships 3,004,166 Total knowledge graph links — C. Natural Language to Cypher Query Translation A key feature of the system is its ability to accept questions written in plain English and automatically generate the corresponding Cypher queries. This is accomplished using Meta’s LLaMA 3 large language model, which runs entirely on a local machine through the open-source llama.cpp framework. Running the model locally ensures low-latency execution and keeps sensitive queries within the user’s environment. To generate a Cypher query, LLaMA 3 is prompted with examples of natural language questions along with their correct Cypher translations. The prompt also includes instructions on how to navigate the structure of the graph schema. When a user enters a question (e.g., ’What are the symptoms of Alzheimer’s disease?’), the system inserts it into the prompt and asks LLaMA 3 to produce a corresponding query. For example, the model may return: MATCH (d:Disease)-[:HAS_SYMPTOM]->(s:Symptom) WHERE toLower(d.name) CONTAINS "alzheimer" RETURN s.name
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Omar & Mohammed / Journal of Applied Science and Technology Trends Vol. 06, No. 02, pp. 342 –357 (2025) 351 This query searches for a disease node whose name contains ’alzheimer’ and follows HAS_SYMPTOM edges to list related symptom names. The system then executes this cypher to retrieve answers. The prompts (such as few-shot examples and schema hints) were carefully designed to help LLaMA 3 generate correct Cypher queries. The model learns how to use the graph’s labels and relationships properly. For example, if a user asks, ’Which drugs treat diabetes?’, LLaMA might respond with a suitable Cypher query: MATCH (d:Drug)-[:TREATS]->(di:Disease) WHERE toLower(di.name) CONTAINS "diabetes" RETURN d.name This queries for drug nodes that have a TREATS edge to a diabetes disease node. By leveraging LLaMA 3 in this way, our system can flexibly handle many phrasing variations without manual mapping rules. D. Model Configuration & Decoding We run a local LLaMA 3.2-3B model in GGUF format (llama- 3.2-3b-instruct-q4_k_m.gguf) via llama.cpp, as shown in Table IV. TABLE IV. MODEL RUNTIME AND DECODING SETTINGS Runtime settings Decoding settings n_ctx = 1024 temperature = 0.2 n_threads = 12 top_p = 0.95 n_gpu_layers = 33 top_k = 40 n_batch = 512 repeat_penalty = 1.1 max_tokens = 80 seed = 42 E. Graph Subset and Versioning We use an iBKH derived subgraph (≈65.8k nodes; ≈3.0M edges) spanning DRUG, DISEASE, SYMPTOM, PATHWAY. IDs are normalized to CURIEs and duplicates collapsed across UMLS/DrugBank/DisGeNET/SIDER/KEGG. Each edge stores provenance/licensing metadata (source, source_version, license, retrieved_at, evidence_pmids/urls) and, when available, temporal fields (valid_from, valid_to). We report coverage as the percentage of evaluated questions whose gold entities/relations are present. F. Query Execution and Reliability After a Cypher query is generated, it is executed on the Neo4j database through the official Neo4j Python driver, which manages the secure connection and returns the results. Instead of restricting the output with a fixed LIMIT (e.g., LIMIT 5), the system retrieves candidate results and evaluates them using standardized retrieval metrics such as Hits@1, Hits@5, and Hits@10. This approach ensures that the system remains responsive while providing a fair assessment of ranking quality across different cutoff levels, rather than depending solely on a fixed number of returned items. Neo4j’s indexing on key node properties, such as name and primary identifiers, also helps speed up lookups as the knowledge graph grows. In cases where the language model generates an incomplete or incorrect query, such as referencing nodes or relationships that do not exist, the system catches the error and either retries with a simpler prompt or informs the user. Together, these steps make sure that queries run quickly, return valid results, and keep the overall experience smooth and reliable for biomedical question-answering. G. User Interface for Query Transparency The system includes a lightweight, cross-platform graphical user interface (GUI) implemented as a web application using the Flask framework in Python, with HTML and Bootstrap for interactive visualization. The interface is designed to make the question-answering process transparent and accessible to users without technical expertise. It consists of three main panels: 1. Input Panel: Where the user can enter a biomedical question in natural language. 2. Query Panel: Which displays the Cypher query generated by the language model, allowing users to verify how their question was interpreted. 3. Results Panel: Which presents the retrieved answers in a clear, readable format, accompanied by a brief natural language explanation generated by the system. By showing both the query and the answer, the GUI promotes user trust and enables validation of the system’s reasoning process. The interface is lightweight enough to run smoothly on standard desktop machines without additional dependencies, making it practical for local deployments in clinical or research settings. Fig. 4 illustrates the overall layout. V. EXPERIMENTAL RESULTS In this section, the proposed system is evaluated based on its ability to translate natural-language biomedical questions into executable Cypher queries over the iBKH knowledge graph. The assessment is conducted end-to-end and focuses on three main aspects: (i) the accuracy of query generation, measured by the proportion of correctly produced Cypher queries; (ii) system efficiency, quantified through total response time from question submission to final answer delivery, covering both query generation and execution; and (iii) the quality of retrieved answers, evaluated using standardized information retrieval metrics including Precision@k, Recall@k, F1@k, Hits@k, and Mean Reciprocal Rank (MRR). For clarity, all these metrics are formally defined in the following subsections, with their corresponding equations, and will be used consistently throughout the results section. Together, these dimensions provide a comprehensive view of both correctness and efficiency across simple, medium, and complex biomedical queries. A. Description of the Experimental Data To evaluate the proposed system, this work used a carefully selected set of biomedical questions designed to test how well the system understands natural language and converts it into Cypher queries for a biomedical knowledge graph. To evaluate the system, a benchmark of 60 biomedical questions was constructed by the authors, guided by the schema and relations in iBKH. The questions were grouped into simple, medium, and complex levels to assess performance across different reasoning requirements. Gold-standard answers were manually prepared for each question to enable quantitative evaluation using Exact Match, Precision@k, Recall@k, F1@k, Hits@k, and MRR. The complete set of 60 questions is available at this link https://drive.google.com/drive/my-drive.
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Omar & Mohammed / Journal of Applied Science and Technology Trends Vol. 06, No. 02, pp. 342 –357 (2025) 352 The dataset contains 60 questions divided into three difficulty levels based on how complex the language is and how deep the biomedical reasoning needs to be: • Level 1: 25 simple questions focusing mostly on easy-to- understand facts, such as symptoms of certain diseases or drugs used to treat specific conditions. • Level 2: 20 medium-level questions that involve more detailed relationships, such as drug interactions and SideEffect. • Level 3: 15 hard questions requiring multi-step reasoning across multiple biomedical concepts or biological pathways, similar to the complexity found in real clinical cases. The set of evaluation questions was designed to span a wide range of common biomedical topics and to reflect clinically relevant query types reported in prior literature. Each question is paired with its corresponding gold standard cypher query and categorized by difficulty level, as summarized in Table V, where three illustrative examples are shown. The questions were derived from publicly available biomedical QA benchmarks and adapted from established knowledge bases (e.g., iBKH schema relations), ensuring both coverage and diversity across diseases, drugs, symptoms, and pathways. TABLE V. QUESTION DIFFICULTY LEVELS AND SAMPLE CYPHER QUERIES Level Definition Example Cypher Query 1 Single-hop question using one relationship What are the symptoms of Alzheimer? MATCH (d:Disease)-[:HAS_SYMPTOM]->(s:Symptom) WHERE toLower(d.name) CONTAINS 'alzheimer' RETURN s.name AS symptom 2 Questions requiring one relationship What are the side effects of drugs used to treat asthma? WITH toLower('asthma') AS disease_name MATCH (d:Disease)<-[:TREATS]-(dr:Drug) WHERE toLower(d.name) CONTAINS disease_name MATCH (dr:Drug)-[:CAUSES]->(se:SideEffect) RETURN DISTINCT se.name AS side_effect, dr.name AS drug 3 Questions requiring two or more relationships What cholesterol medications cause side effects, and what are some of those effects? WITH toLower('cholesterol') AS disease_name MATCH (d:Disease)<-[:TREATS]-(dr:Drug) WHERE toLower(d.name) CONTAINS disease_name MATCH (dr:Drug)-[:CAUSES]->(se:SideEffect) RETURN DISTINCT dr.name AS drug, se.name AS side_effect Fig. 4. Graphical User Interface of the Biomedical Knowledge Explorer System B. Quantitative Evaluation The performance of the proposed system is evaluated on the iBKH knowledge graph using Exact Match, Precision@k, Recall@k, F1@k, Hits@k, MRR, and total response time across simple, medium, and complex biomedical queries. These metrics were measured for each difficulty level (simple, medium, and complex) to see how performance changes as questions become more challenging. TABLE VI. QUERY GENERATION EXACT MATCH BY DIFFICULTY LEVEL
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Omar & Mohammed / Journal of Applied Science and Technology Trends Vol. 06, No. 02, pp. 342 –357 (2025) 353 Difficulty Level Total Questions Correct Queries Cypher Exact Match (EM) (%) Simple 25 24 96% Medium 20 19 95% Complex 15 13 86.7% avg 92.6% To better understand the quality of the responses returned by the system, this work examined real examples. One of the test questions was "What drugs are used to treat breast cancer?" As shown in Fig. 4, the system was able to understand the question, generate the correct Cypher query using the TREATS relationship, and retrieve the right information from the biomedical knowledge graph. It returned a list of five drugs, including Leuprolide, Goserelin, and Trastuzumab, which are known treatments for breast cancer. This result shows that the system is able to connect the question to the right part of the graph and provide accurate and helpful answers, even for medical questions that involve specific treatments. 1) Exact Match of Query Generation As shown in Table VI, how often the system generated the correct query for each difficulty level. Here, accuracy is defined as the percentage of questions for which the system’s generated Cypher query matched the expected query. It is calculated using the Eq. (1) Exact Match (%) = Correct Queries Total Questions × 100 (1) These findings highlight the purpose of this experiment to test whether the framework can reliably map natural language to Cypher across varying levels of complexity. The graceful drop from 96% on simple to 86.7% on complex queries indicates that the system is robust for straightforward questions but still challenged by multi-hop reasoning. This points to clear opportunities for improvement, such as synonym expansion, constrained decoding, or enhanced error handling for multi-step queries. 2) Latency Table VII reports the average latency per difficulty level and decomposes it into query generation and Neo4j execution. The total response time is computed as in Eq. (2). Execution time is effectively constant across all levels ( ≈0.04–0.05 s), so variation in total latency is dominated by query generation. As difficulty increases, the mean total time rises from 5.12 s (simple) to 5.75 s (medium) and 7.35 s (complex). Dispersion (Std. Dev.) grows with task complexity 0.72 s (simple), 0.32 s (medium), 2.09 s (complex) reflecting more variable planning and reasoning needed to assemble correct Cypher for harder questions. Pooled over all questions, the overall mean is 6.07 s with an overall SD of 1.38 s, keeping latencies within single-digit seconds and practical for interactive, real-world use. 𝑇total = 𝑇gen + 𝑇exec (2) Standard Deviation (SD). Unless otherwise stated, SD is the unbiased sample standard deviation computed over per-question total times within each difficulty group 𝐺 with 𝑛𝐺 questions: 𝑇‾𝐺= 1 𝑛𝐺 ∑𝑇total (𝑖) 𝑛𝐺 𝑖=1 , 𝑠𝐺= √ 1  𝑛𝐺−1  ∑ 𝑛𝐺 𝑖=1 (𝑇total (𝑖) −𝑇‾𝐺) 2 . (3) (4) Overall values are computed by pooling all questions across levels, with 𝑁= ∑𝑛𝐺 𝐺 : 𝑇‾overall = 1 𝑁∑𝑇total (𝑖) 𝑁 𝑖=1 , 𝑠overall = √ 1  𝑁−1  ∑ 𝑁 𝑖=1 (𝑇total (𝑖) −𝑇‾overall) 2 . (5) (6) TABLE VII. AVERAGE LATENCY (S) BY DIFFICULTY LEVEL (SD = STANDARD DEVIATION) Difficulty Level Query Generation (s) Query Execution (s) Total Time (s) Std. Dev. (s) Simple 5.09 0.03 5.12 0.72 Medium 5.69 0.01 5.75 0.32 Complex 6.94 0.4 7.35 2.09 Overall 5.9 0.15 6.07 1.38 3) Answer-Level Evaluation Metrics As shown in Table VIII, the quality of the returned items is evaluated using five standardized metrics: Precision@k, Recall@k, F1@k, Hits@k, and MRR. Precision@k measures the proportion of correct answers among the top-𝑘 retrieved items Eq. (7), while Recall@k quantifies the fraction of gold-standard answers covered within the top-𝑘 results Eq. (8). F1@k combines both aspects through the harmonic mean Eq. (9). Hits@k reports whether at least one correct answer appears in the top-𝑘 Eq. (10, 11), and MRR captures how early the first correct answer is retrieved in the ranking Eq. (12). Together, these metrics provide a comprehensive view of both the correctness and completeness of retrieval, as well as the ranking quality across simple, medium and complex queries. Precision@𝑘= |{ relevant ∩retrieved@𝑘 }| 𝑘 (7) Precision@k measures the fraction of the top-𝑘 retrieved items that are correct (i.e., appear in the gold set). For example,
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Omar & Mohammed / Journal of Applied Science and Technology Trends Vol. 06, No. 02, pp. 342 –357 (2025) 354 P@1 refers to the accuracy of the very first retrieved item, P@5 evaluates correctness within the top three results, and P@10 considers the top ten. Higher values indicate that relevant items tend to appear early in the ranked list. TABLE VIII. EVALUATION METRICS ACROSS LEVELS Simple Medium Complex Overall P@1 100 95 86.67 93.8 P@5 100 95 93.33 96.11 P@10 100 95 88 94.33 R@1 88 90 86.67 88.22 R@5 90.65 89.67 86.67 88.9 R@10 89.60 89 83.33 87.31 F1@1 88 90 86.67 88.22 F1@5 93.20 91.25 88.67 91.03 F1@10 93.28 91.39 85.24 89.97 Hits@k 100 95 93 96.1 MRR 100 95 88 94.4 Recall@𝑘= |{ relevant∩retrieved@𝑘 }| |{ relevant }| (8) Recall@k measures the proportion of all relevant items (in the gold set) that are successfully retrieved within the top- 𝑘 positions. For instance, R@5 indicates how many of the expected answers are covered by the top three results. This metric is particularly important when the gold answer set is larger than 𝑘. F1@𝑘= 2 ⋅Precision@𝑘⋅Recall@𝑘 Precision@𝑘+ Recall@𝑘 (9) F1@k is the harmonic mean of Precision@k and Recall@k. It balances the trade-off between retrieving only relevant items (precision) and covering as many relevant items as possible (recall). For example, F1@10 reflects the combined quality of the system when retrieving the top ten results. Hits@𝑘= {1 if { relevant ∩retrieved@𝑘 } ≠⌀ 0 otherwise (10) Hits@k is a binary measure at the query level: it equals 1 if at least one correct answer is present among the top-𝑘 retrieved items, and 0 otherwise. For example, Hits@5 reports the percentage of queries where the system was able to “hit” at least one correct answer in the top five results. Hits@𝑘= 1 |𝑄| ∑𝟏 𝑞∈𝑄 ({ relevant𝑞 ∩retrieved𝑞@𝑘 } ≠⌀) (11) When averaged across a set of queries 𝑄, Hits@k gives the proportion of queries for which at least one relevant item is returned in the top-𝑘 results. This measure is less sensitive to ranking quality but emphasizes coverage across queries. MRR = 1 |𝑄| ∑ 1 rank𝑞 𝑞∈𝑄 (12) Mean Reciprocal Rank (MRR) averages the reciprocal of the rank of the first correct answer for each query 𝑞∈𝑄. For example, if the first correct answer appears in the top position, the reciprocal rank is 1/1 = 1.0; if it appears at rank 5, the contribution is 1/5 = 0.2. MRR therefore rewards systems that retrieve correct answers as early as possible. According to the results in Table VIII, this work reports standardized metrics, including Precision, Recall, F1, Hits@k, and MRR at different cutoffs (𝑘= 1,5,10). Precision@k captures the proportion of retrieved items among the top-𝑘 that are correct, while Recall@k measures the coverage of gold-standard answers within the same cutoff. F1@k balances both aspects. Hits@k reflects whether at least one correct answer appears in the top-𝑘, and MRR evaluates how early the first correct answer is retrieved. For simple questions ( 𝑁= 25 ), the performance is consistently near perfect, with P @ 10 = 100%, R @ 10 (89. 6%) and F1 @ 10(93. 3%), along with Hits @ 10 = 100% and MRR = 100%. For medium questions (𝑁= 20), P @ 10 = 95%, R @ 10 (89%) and F1 @ 10 (91. 4%), with a strong ranking quality reflected in Hits@10 = 95% and MRR = 95%. For complex queries (𝑁= 15), the performance remains robust but slightly lower, with P @ 10 (93. 3%), R @ 10 (88%) and F1 @ 10 (90. 2%), alongside Hits @ 10 (93. 3%) and MRR 93. 3%. In general, at all levels, the system achieves P @ 10 (96. 1%), R @ 10 (88. 9%) and F1 @ 10 (91. 6%), with Hits @ 10 (96. 1%) and MRR 96. 1%. These results indicate that the system not only retrieves nearly all expected answers but also ranks them highly, ensuring both completeness and correctness. This level of reliability is particularly valuable in biomedical applications where precision and trustworthiness are critical. The main purpose of this experiment was to assess not only whether queries execute, but whether the returned results are both correct and complete. The precision consistently above 95% confirms that almost all the items retrieved are clinically valid, while Hits@5 near 95% shows that the system usually returns close to the expected five answers per question. Together, these metrics demonstrate that knowledge graph grounding effectively minimizes hallucinations and ensures trustworthy biomedical output. We evaluated performance differences across difficulty levels using a two-sample t-test, which revealed statistically significant differences. Error analysis indicates that failures in complex queries are mainly due to missing relation hops, whereas errors in medium-level queries are mostly caused by syntax mismatches. These findings highlight the challenges associated with query complexity and provide insights for targeted improvements. C. Qualitative Evaluation In addition to quantitative metrics, the system’s outputs were evaluated for contextual accuracy and alignment with the structure of the knowledge graph. Two plain English questions were selected and for each, the generated Cypher query, the Neo4j output, and the visual graph representation were reviewed to verify that the answers reflected valid relationships in the graph.
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Omar & Mohammed / Journal of Applied Science and Technology Trends Vol. 06, No. 02, pp. 342 –357 (2025) 355 For the question ’What are the symptoms of brain cancer?’ The system generated a Cypher query that correctly followed the HAS_SYMPTOM relationship from disease nodes to symptoms nodes, filtering by the specified disease name. The results retrieved included terms such as aphasia (Broca, Wernicke), anomia, agraphia, agnosia, amnesia (including retrograde amnesia), olfaction disorders, and apnea symptoms consistent with established neurological manifestations of brain tumors. In Neo4j, the data formed a clear center-and-spoke pattern, with brain cancer at the center and its associated nodes of symptoms radiating outward, as shown in Fig. 5. Cypher: WITH toLower("brain cancer") AS disease_name MATCH (d:Disease)-[:HAS_SYMPTOM]->(s:Symptom) WHERE toLower(d.name) CONTAINS disease_name RETURN DISTINCT s,d LIMIT 10; Fig. 5. Graphical user interface displaying Cypher query and results for breast cancer treatment A second query, What are the side effects of insulin?, produced a Cypher statement starting from the drug node for insulin and traversing the CAUSES relationship to the connected SideEffect nodes. The resulting list included dizziness, diarrhea, cough, back pain, weakness, rash/dermatitis, and hypersensitivity side effects well documented in insulin safety profiles. In the visual representation in Fig. 6, insulin appeared centrally connected to these side effect nodes, further strengthening the correctness of the relationship mapping of the system. Cypher: WITH toLower("insulin") AS drug_name MATCH (d:Drug)-[:CAUSES]->(se:SideEffect) WHERE toLower(d.name) CONTAINS drug_name RETURN se,d LIMIT 20; Fig. 6. Knowledge Graph Visualization of Insulin and Its Related Side Effects These examples demonstrate the system’s ability to interpret plain English biomedical questions, generate correct Cypher queries, and return results that are clinically plausible, easy to interpret, and directly traceable to specific graph relationships. This supports both the accuracy and the transparency of the proposed approach. To illustrate how biomedical questions can be translated into graph queries, consider the natural language question: ’What are the side effects of drugs that treat epilepsy?’. The following Cypher query demonstrates how this question is mapped in the knowledge graph, where diseases are connected to drugs through the TREATS relation and drugs are connected to side effects through the CAUSES relation. Cypher: MATCH (d:Disease)<-[:TREATS]-(dr:Drug) WHERE toLower(d.name) CONTAINS "epilepsy" MATCH (dr:Drug)-[:CAUSES]->(se:SideEffect) RETURN DISTINCT se,d,dr limit 10
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Omar & Mohammed / Journal of Applied Science and Technology Trends Vol. 06, No. 02, pp. 342 –357 (2025) 356 Fig. 7. Side effects of drugs that treat epilepsy Executing this query returns drugs associated with epilepsy and their corresponding side effects. For example, the query identifies Pregabalin as a treatment for epilepsy and retrieves multiple side effects such as anxiety, arrhythmia, gastrointestinal pain, and others. The visualization of the graph in Fig. 7, highlights this pattern, where the drug node is linked to epilepsy via TREATS and to several side effect nodes via CAUSES, providing an interpretable biomedical knowledge structure. VI. DISCUSSION Under a unified evaluation protocol reporting P@k/R@k/F1@k, Hits@k, and MRR at 𝑘∈{1,5,10} , the pipeline exhibits consistent end-to-end behavior across the three difficulty tiers. The 𝑘-ablation shows strong early precision (high P@1 and elevated MRR), while recall increases with larger 𝑘, indicating that correct answers typically surface near the top yet persist deeper in the ranked slate. At the query-generation level, exact-match (EM) is high for simple questions (e.g., ∼96%) and lower for complex, multi-hop questions (e.g., ∼86.7%), which aligns with increased compositionality. End-to-end latency (mean total ≈6.07 s from question to final answer) remains within interactive bounds on our local setup. Practically, a fully offline deployment avoids internet connectivity, API keys, and external data transfer, strengthening privacy, compliance, and reproducibility in biomedical contexts. Several challenges qualify these findings. First, the comparability challenge: to our knowledge, no prior study evaluates iBKH using the same metric suite, making cross-paper numeric comparisons not “apples to apples.” We therefore interpret the results as controlled evidence about this pipeline under a single, consistent protocol rather than as a cross-study leaderboard. Beyond comparability, performance may vary with other biomedical knowledge graphs; the current iBKH snapshot is static, limiting real-time updates; and scaling to larger or dynamically refreshed graphs can introduce latency and consistency trade-offs. Error analysis shows that residual failures concentrate in complex, multi-hop queries where missing relation hops or brittle name-based matching (synonyms, abbreviations, homonyms) lead to partial answers. These observations motivate concept- level normalization via biomedical identifiers (e.g., UMLS, SNOMED, RxNorm) with synonym expansion, as well as schema-constrained query generation and path-guided decoding to better satisfy multi-hop constraints. Finally, correctness is assessed primarily at the answer level (Hits@k, MRR, precision/recall) and does not yet include full semantic- equivalence checks across alternative Cypher queries, which may overlook cases where different queries yield the same correct results. Complementing template EM with result-set equivalence checks, expanding the metric suite (e.g., nDCG/MAP) with per- question 95% bootstrap confidence intervals, and supporting incremental graph updates with distributed storage are promising steps to enhance robustness, scalability, and external validity. VII. CONCLUSION This study introduced a hybrid biomedical question answering framework that couples the LLaMA-3B language model with a Neo4j-based iBKH knowledge graph to enable the automatic generation of executable Cypher queries and to deliver transparent, evidence-grounded answers through justification subgraphs. Evaluation in simple, medium, and complex queries demonstrated consistently high performance, with strong precision, recall, F1, Hits@k, and MRR values, while maintaining low latency suitable for interactive biomedical applications. Beyond quantitative performance, the proposed system provides a reproducible and privacy-preserving solution by operating fully offline, a property of particular importance in sensitive clinical and research contexts. However, certain limitations remain. The reliance on a static iBKH snapshot constrains coverage and adaptability, recall is lower for complex multi-hop reasoning, and the absence of canonical entity normalization (e.g., UMLS, SNOMED, RxNorm) may reduce semantic robustness. Future research will therefore focus on integrating standardized biomedical entity normalization, enabling dynamic and incremental knowledge graph updates, and leveraging domain-adapted or fine-tuned biomedical LLMs. These directions are expected to further strengthen the robustness, scalability, and applicability of the framework in real- world biomedical and clinical environments. REFERENCES [1] S. Bubeck, V. Chandrasekaran, R. Eldan, J. Gehrke, E. Horvitz, E. Kamar, P. Lee, Y. T. Lee, Y. Li, S. Lundberg, et al., “Sparks of artificial general intelligence: Early experiments with gpt-4,” arXiv preprint arXiv:2303.12712, 2023. [2] J. Achiam, S. Adler, S. Agarwal, L. Ahmad, I. Akkaya, F. L. Aleman, D. Almeida, J. Altenschmidt, S. Altman, S. 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DALK: Dynamic Co-Augmentation of LLMs and KG to answer Alzheimer’s Disease Questions with Scientific Literature Dawei Li1*, Shu Yang2∗, Zhen Tan1, Jae Young Baik2, Sunkwon Yun3, Joseph Lee2, Aaron Chacko2, Bojian Hou2, Duy Duong-Tran2,4, Ying Ding5, Huan Liu1†, Li Shen2†, Tianlong Chen3† 1School of Computing, and Augmented Intelligence, Arizona State University 2Department of Biostatistics, Epidemiology and Informatics, University of Pennsylvania Perelman School of Medicine 3Department of Computer Science, The University of North Carolina at Chapel Hill 4Department of Mathematics, United States Naval Academy 5School of Information, The University of Texas at Austin, Austin Abstract Recent advancements in large language mod- els (LLMs) have achieved promising perfor- mances across various applications. Nonethe- less, the ongoing challenge of integrating long-tail knowledge continues to impede the seamless adoption of LLMs in specialized do- mains. In this work, we introduce DALK, a.k.a. Dynamic Co-Augmentation of LLMs and KG, to address this limitation and demonstrate its ability on studying Alzheimer’s Disease (AD), a specialized sub-field in biomedicine and a global health priority. With a synergized frame- work of LLM and KG mutually enhancing each other, we first leverage LLM to construct an evolving AD-specific knowledge graph (KG) sourced from AD-related scientific literature, and then we utilize a coarse-to-fine sampling method with a novel self-aware knowledge re- trieval approach to select appropriate knowl- edge from the KG to augment LLM infer- ence capabilities. The experimental results, conducted on our constructed AD question answering (ADQA) benchmark, underscore the efficacy of DALK. Additionally, we per- form a series of detailed analyses that can of- fer valuable insights and guidelines for the emerging topic of mutually enhancing KG and LLM. We will release the code and data at https://github.com/David-Li0406/DALK. 1 Introduction Alzheimer’s Disease (AD) is a neurodegenerative disorder characterized by progressive declines in cognitive and functional status over a span of decades (Report, 2023). However, current AD ther- apy developments are facing critical challenges due to the lack of knowledge and understanding of the underlying etiological mechanisms of the dis- ease. Although scientific literature and dedicated biomedical databases could supply rich sources of * Equal Constributions † Corresponding authors AD knowledge, manual review of relevant informa- tion is impossible due to the large volume. As large language models (LLMs) (Brown et al., 2020; Zhang et al., 2022; Anil et al., 2023; Touvron et al., 2023) with chain-of-thought (CoT)-based prompting (Wei et al., 2022; Wang et al., 2022; Tong et al., 2023; Yao et al., 2023; Besta et al., 2023) demonstrate strong language capabilities across various tasks, there have been attempts to leverage LLMs-based systems in general biomedi- cal and AD-related applications (Mao et al., 2023; Li et al., 2023c; Yan et al., 2024; Feng et al., 2023). However, while the LLMs have shown promising performances in many general tasks, recent studies revealed LLMs’ limitations in long-tail (Kandpal et al., 2023) and domain-specific (Li et al., 2023b, 2024) knowledge, thereby significantly impeding their adaptations in vertical fields such as AD. To deal with this issue, the most common strategies are retrieval augmented generation (RAG) and domain- specific LLMs training. Nevertheless, directly applying these strategies in the context like AD would still suffer from sev- eral issues. First, Data Quality: As in many biomedical fields, scientific literature composes the largest publicly available corpus source in AD. Yet, the dense and information-overloaded nature of scientific literature, when combined with auto- matic retrieval methods, can lead to the retrieval of irrelevant and noisy information. Previous re- search has shown that noisy and irrelevant corpora can significantly undermine the performance of LLMs (Yu et al., 2023; Chen et al., 2024; Wu et al., 2024). Second, Efficiency & Scale Issues: Being an critical field of research, the knowledge of AD is rapidly evolving with scientific advancements at a remarkable pace and scale. However, retraining a domain-specific LLM or updating certain knowl- edge in it demands substantial computational re- sources (Hu et al., 2021; Ovadia et al., 2023; Zhang et al., 2024). This efficiency issue would also limit arXiv:2405.04819v1 [cs.CL] 8 May 2024
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the sizes of domain-specific LLMs, consequently affecting their performances. To tackle these limitations, here we propose a Dynamic Co-Augmentation of LLMs and KG (DALK) framework that facilitates mutual benefits between LLMs and knowledge graphs (KG) for the AD domain. Initially, our framework addresses the data quality challenge by extracting more struc- tural and accurate knowledge from unstructured and dense scientific literature and constructing a domain-specific knowledge graph tailored to AD. We employ two widely utilized knowledge graph construction methods, namely pair-wise construc- tion (Carta et al., 2023; Wadhwa et al., 2023) and generative construction (Han et al., 2023; Bi et al., 2024), to comprehensively assess their impact on knowledge graph quality. Then, we adopt a coarse- to-fine sampling method with a novel self-aware knowledge retrieval approach to select appropri- ate knowledge from the knowledge graph and thus further address the data quality problem. Notably, the tuning-free nature of our framework signifi- cantly enhances efficiency and facilitates its ap- plication in large-scale and API-based language models (OpenAI, 2022). In the evaluation sec- tion, we derive an Alzheimer’s Disease question answering (ADQA) benchmark from existing gen- eral medical QA datasets with millions of sam- ples filtered by a curated keyword list and self- sampling of LLMs. Our extensive experiment on ADQA demonstrates the effectiveness of our frame- work in domain-specific applications compared with general biomedical LLMs and retrieval aug- mented models. Further evaluation and analysis provide valuable insights into constructing high- quality knowledge graphs and sampling accurate knowledge from them. In summary, our contribution in this work can be summarized as follows: • We identify the constraints of the current meth- ods for LLMs in domain-specific areas like AD and introduce DALK, a co-augmentation framework of the LLM and KG to address these issues. • We build AD-specific KG and QA benchmark. Through extensive comparisons with other methods, we showcase the effectiveness of DALK. • We delve into a comprehensive analysis of our proposed method and provide valuable insights and guidance on how to construct a high-quality KG and sample accurate knowl- edge from it. 2 Related Work The interplay between LLMs and KGs KGs (Miller, 1995; Speer et al., 2017; Vrandeˇci´c and Krötzsch, 2014) serve as structured represen- tations of factual knowledge, typically expressed as (head, relation, tail) triples. Their structured, factual, and interpretable nature renders them ex- cellent complements to parametric language mod- els (Pan et al., 2024). Recently, with the rise of large language models (LLMs), numerous studies have delved into exploring the synergy between LLMs and KGs for various purposes (Pan et al., 2024; Tan et al., 2024). There are a lot of efforts in conducting knowledge graph construction (Carta et al., 2023; Wadhwa et al., 2023; Han et al., 2023; Bi et al., 2024; Datta et al., 2024), completion (Wei et al., 2023; Zhang et al., 2023b; Li et al., 2024) with the aid of LLMs. Conversely, other works aim to enhance LLMs by integrating knowledge sam- pled from KGs during both training (Tang et al., 2023; Luo et al., 2024; Dernbach et al., 2024; Rangel et al., 2024) and inference (Kim et al., 2023; Wen et al., 2023; Jiang et al., 2023; Sun et al., 2023a) times. Our work distinguishes itself by proposing a co-augmentation framework for LLMs and KGs, facilitating their mutual enhancement, and applying it to the domain of AD. LLMs and KGs for AD research LLMs and KGs have both been applied to Alzheimer’s Dis- ease research in previous studies. Pre-trained lan- guage models are utilized to work on AD de- tection and many other related tasks based on speech recordings and transcripts (Balagopalan et al., 2020; Agbavor and Liang, 2022), electronic health records (EHRs) (Mao et al., 2023; Li et al., 2023c; Yan et al., 2024), and tabular data (Feng et al., 2023). KGs have been widely used in biomedical research, yet only a few are specifically for AD research (Romano et al., 2023; Pu et al., 2023; Hsieh et al., 2023; Nian et al., 2022; Daluwa- tumulle et al., 2023). These KGs were generally constructed from a variety of information derived from heterogeneous biomedical databases (e.g. for genes, drugs, pathways, etc.) or scientific literature related to AD. Despite the aforementioned efforts for LLMs and KGs in AD research, no prior study has explored using LLM to augment AD-KG, or
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vice versa, let alone the potential for mutual en- hancement between the two as we propose here. 3 Our Methodology This section elaborates on our dynamic co- augmentation framework of LLMs and KG. Sec- tion 3.1 presents the details of augmenting an AD- specific evolving KG with LLMs and literature corpus in a time-slicing fashion (i.e. year by year). Following it, Section 3.2 describes the process of sampling appropriate knowledge from the evolving KG to enhance LLMs’ reasoning. Figure 1 illus- trates an overall pipeline of our method DALK. KGpair KGgen #Corpus 9,764 9,764 #Nodes 13,509 20,545 #Relations 3,952 3,651 #Triples 171,431 53,585 Table 1: Detailed statistics about our augmented knowl- edge graph. 3.1 LLMs for KG Corpus Collection To create an AD-specific knowledge graph, we follow (Pu et al., 2023) and use the AD corpus collected by a domain expert Professor Colin Masters at the University of Mel- bourne who discovered amyloid proteins being the potential cause of AD (Masters et al., 1985). The corpus is based on his extensive bibliography of representative AD-related papers and consists of more than 16K PMID (PubMed ID)-indexed arti- cles from 1977 to 2021. For our study, we focus on the papers since 2011 which reflect the most recent knowledge in the field and get 9,764 articles. Entity Recognition In order to identify knowl- edge at the proper granularity level for AD, we extract relevant entities from the corpus by utiliz- ing the PubTator Central (PTC) (Wei et al., 2013) developed and continuously maintained by NCBI. PTC is a widely-used tool to provide state-of-the- art annotations of biomedical concepts for PubMed abstracts and full-text articles, and it supports six bioconcept types including genes, diseases, chem- icals, mutations, species and cell lines. We apply PTC to the abstracts of all our AD papers and ob- tain the relevant named entities which will serve as nodes in the knowledge graph. Relation Extraction To build an accurate and high-quality knowledge graph on AD, we aim to as- sign a specific relation type between the two related entities. Through a comprehensive survey of rela- tion extraction methods for knowledge graph con- struction, we categorize current approaches with LLMs into two main groups: (a). Pair-wised Rela- tion Extraction (Carta et al., 2023; Wadhwa et al., 2023) aims to prompt the LLMs to describe the relationship between any two entities in a segment of text. (b). Generative Relation Extraction (Han et al., 2023; Bi et al., 2024; Datta et al., 2024), where LLMs directly output all related entity pairs and their corresponding relationships. As shown in Figure 2, we incorporate both of these rela- tion extraction methods into our knowledge graph augmentation process to provide a comprehensive comparison between them. We denote the result- ing knowledge graphs from these approaches as KGpair and KGgen respectively. Table 1 presents the detailed statistics about our augmented knowledge graph, including the num- ber of corpora we used, and the number of nodes, relations and triples in KGpair and KGgen. 3.2 KG for LLMs In this section, we begin by outlining our process for sampling coarse-grained augmented knowledge from our evolving knowledge graph (Section 3.2.1). Subsequently, we delve into detail regarding our self-aware knowledge retrieval method, which aims to filter out noise and retrieve the most pertinent knowledge to provide to the LLM (Section 3.2.2). 3.2.1 Coarse-grained Knowledge Sample Given a question query Q, we first construct a prompt and ask LLMs to extract all the domain- specific entities E = {e1, e2, ...} from it. Af- terward, we adhere to the methodology proposed by Wen et al. (2023) and execute a similarity-based entity linking process to connect all entities within E to the entity structure in our knowledge graph G. Specifically, we employ a semantic similarity model (Reimers and Gurevych, 2019) to encode all entities in G and E into dense embeddings, denoted as HG and HE, respectively. Subsequently, utiliz- ing cosine similarity, we establish links between each entity in E and its nearest neighbor entity in G. This procedure yields an initial entity set EG for the subsequent knowledge sampling step. To build an evidence sub-graph to boost LLMs’ reasoning process, we follow the previous
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… Successful application of AD preventative approaches may hinge on an accurate and comprehensive view of comorbidities, including cardiovascular disease, diabetes, and head trauma. Literature Corpus LLMs for KG KG for LLMs Head Relation Tail Diabetes AD Head trauma … … … risk factor AD risk factor Extracted Triplets Evolving KG Evolving KG AD Cognition level Down’s syndrome Head trauma Diabetes Selected Knowledge LLMs LLMs Extract Process Augment Q: Following are predispositions to Alzheimer's disease except ___. A). Down’s syndrome B). Head trauma C). Smoking D). Low education group Answer Rerank Sample AD Cognition level Down’s syndrome Head trauma Diabetes Subgraph Sampling Genetic factors Lifestyle Input Q: Following are predispositions to Alzheimer's disease except ___. A). Down’s syndrome B). Head trauma C). Smoking D). Low education group Question Input Figure 1: The overview pipeline of DALK. We first extract structural knowledge from unstructured corpora and construct a domain-specific knowledge graph tailored to AD (Section 3.1). Then, we utilize a coarse-to-fine sampling method with a novel self-aware knowledge retrieval approach to select appropriate knowledge from the knowledge graph (Section 3.2). … Successful application of AD preventative approaches… , including cardiovascular disease, diabetes, and head trauma. … Successful application of AD preventative approaches… , including cardiovascular disease, diabetes, and head trauma. Step1: Entity Recongnition Step2: Relation Extraction Pair-wised Relation Extraction Generative Relation Extraction … Reading the following abstract: [Abstract] Question: What is the relationship between AD and cardiovascular disease? Question: What is the relationship between AD and diabetes? Reading the following abstract: [Abstract] Here are all the biomedicine-related entities: [Entities] Question: Please extract all the valid relationship between the provided entities. Figure 2: The detailed process of AD-specific KG con- struction. study (Wen et al., 2023) and consider the following two kinds of explorations in our AD-KG: Path-based Exploration entails the extraction of a sub-graph from G to encompass all entities within EG. The process unfolds as follows: (a) Begin by selecting one node from e0 Q as the initial node, denoted as e1, and place the remaining nodes into a candidate node set, Ecand. Explore at most k hops from e1 to identify the subsequent node, e2, where e1 ∈Ecand. If e2 is successfully reached within k hops, update the start node to e2 and remove e2 from Ecand. In the event e2 cannot be found within k hops, concatenate the segment paths acquired thus far and store them in Gpath Q . Subsequently, choose another node e′ 1 from Vcand as the new start node, and eliminate both the original start node e1 and the current node e2 from Ecand. (b) Verify if Ecand is empty. If not, repeat step (a) to identify the next segment of the path. If Ecand is empty, combine all segments to construct a set of sub- graphs and place them into Gpath Q . Neighbor-based Exploration endeavors to aug- ment the evidence relevant to the query within GQ. This process consists of two steps: (a) Initially, expand each node e within EG by 1-hop to incor- porate their neighbors e′, thus appending triples (e, r, e′) to Gnei Q . (b) Then assess whether each e′ exhibits semantic relevance to the query. If affir- mative, further expand the 1-hop neighbors of e′, consequently adding triples (enei, r′, e′) to Gnei Q . After obtaining the two sub-graphs Gpath Q and Gnei Q , we perform post-processing to further prune redundant information in sub-graphs and prompt LLMs to describe the structure of each sub-graph. 3.2.2 Self-aware Knowledge Retrieval In our initial experiment, we noticed the coarse- grained knowledge sampled with the above- mentioned approaches still contained redundant and irrelevant information. This issue of noise is a common challenge encountered in automatically- constructed knowledge graphs (Fang et al., 2021; Zhang et al., 2020; Li et al., 2022; Bi et al., 2024).
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Moreover, many recent works (Yu et al., 2023; Li et al., 2023d; Chen et al., 2024; Wu et al., 2024) have demonstrated LLMs can indeed be influenced by such noisy information. To address this chal- lenge, we borrow insights from the recent self- powered LLMs (Wang et al., 2022; Pan et al., 2023; Li et al., 2023a; Yuan et al., 2024; Tong et al., 2024) and propose a self-aware knowledge retrieval method to leverage LLMs’ ranking capability (Sun et al., 2023b; Ma et al., 2023) to filter out noisy information. In particular, we directly prompt the LLM to rerank the sampled knowledge and only retrieve top k triples to provide for itself in the final-round inference. Given the question Q and either the path- based or neighbor-based sub-graph GQ, we create prompt pself by filling the pre-defined template: pself = Templateself(Q, GQ, k). (1) Then, we use pself as the input to prompt the LLM to obtain the self-retrieved knowledge: Gself Q = LLM(pself), (2) Finally, we provide the question Q and fine-grained knowledge Gself Q to the LLM for reasoning and get the predicted answer a in two steps: pinference = Templateinference(Q, Gself Q ), (3) a = LLM(pinference). (4) We provide detailed examples in Appendix A and B to demonstrate the input and output in our DALK. 4 Main Experiment 4.1 ADQA Benchmark For performance evaluation, we consider four widely-used medical QA datasets spanning diverse biomedical topics (Jin et al., 2021; Pal et al., 2022; Hendrycks et al., 2021; Peñas et al., 2013) and de- rive an AD-specific QA dataset from them. The four medical QA datasets are all multiple-choice based and include: 1) MedQA (Jin et al., 2021) consisting of US Medical Licensing Examina- tion (USMLE)-style questions, 2) MedMCQA (Pal et al., 2022) containing medical school entrance exam questions from India, 3) MMLU (Hendrycks et al., 2021) consisting of diverse biomedical and clinical questions from various sources, 4) QA4MRE (Peñas et al., 2013) containing a sub- set of questions for AD derived from PubMed and Medline. In order to extract from the med- ical QA datasets a subset of samples related to AD for our evaluation, we referred to NIH’s Com- mon Alzheimer’s and Related Dementias Research Ontology (CADRO) 1. Jointly developed by the National Institute on Aging and the Alzheimer’s Association, CADRO is a three-tiered classifica- tion system with eight main categories and a dozen sub-categories for AD and related dementia, and it contains common terminologies or keywords used in the field. We derived from the CADRO a list of AD-related keywords most relevant to the medical QA datasets: <Aging, Alzheimer, Amyloid beta, APOE, Dementia, Lipoprotein, Microglia>. Then, we searched against each medical QA dataset for matches with these keywords to find putative QA samples, then further asked GPT-3.5-turbo to judge for each putative sample whether the question is indeed related to AD or not. Finally, we filtered out a subset of such samples that are considered highly relevant to AD to conduct our evaluation (number of samples in each dataset is shown in Table 2). More details about ADQA can be found in Appendix C. 4.2 Experiment Settings We apply our framework with OpenAI GPT-3.5- turbo models (OpenAI, 2022). We also include the following baseline methods for comparison: Biomedical LLMs Both ChatDoctor (Yunxiang et al., 2023) and Med-Alpaca (Shu et al., 2023) are fine-tuned versions of LLaMA (Touvron et al.) on biomedical corpora. Compared with them, Meditron (Chen et al., 2023) is built on LLaMA- 2 (Touvron et al., 2023) and extends its pretrain- ing on a comprehensively curated medical cor- pus. BiomedGPT (Zhang et al., 2023a) is also based on LLaMA-2 and pioneer as the first open- source and generalist visual language AI for di- verse biomedical tasks. Biomistral (Labrak et al., 2024) is an open-source LLM crafted specifically for the biomedical domain, optimized for effi- ciency through quantization and model merging techniques. Retrieval-Augmented LLMs Furthermore, we also compare our method with several represen- tative retrieval-augmented LLMs in the biomed- ical domain. Almanac (Zakka et al., 2024) is a novel approach utilizing OpenAI’s GPT model in- 1https://iadrp.nia.nih.gov/about/cadro
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MedQA MedMCQA MMLU QA4MRE AVG Biomedical LLMs ChatDoctor (Yunxiang et al., 2023) 25.7 36.4 46.9 51.4 40.1 Med-Alpaca (Shu et al., 2023) 41.4 42.8 44.9 57.1 46.5 BiomedGPT (Zhang et al., 2023a) 38.8 41.9 48.9 42.6 43.1 Meditron (Chen et al., 2023) 27.6 31.4 36.7 25.7 30.4 Biomistral (Labrak et al., 2024) 44.7 49.5 53.1 68.6 54.0 Retrieval-augmented LLMs GPT-3.5-turbo w/ Ada (OpenAI, 2024) 57.2 65.7 83.7 62.9 67.4 Almanac (Zakka et al., 2024) 48.0 69.5 71.4 60.0 62.2 Clinfo.ai (Lozano et al., 2023) 54.3 77.0 81.3 67.7 70.1 Clinfo.ai w/o PubMed API 49.3 68.6 79.6 74.3 67.9 GPT-3.5-turbo 50.0 71.9 83.6 62.9 67.1 DALK 57.9 75.2 85.4 71.4 72.6 Table 2: Experiment results on our constructed ADQA benchmark. The sample size is indicated after each dataset name. The best results of each metric are in bold and the second-best results are underlined. The “AVG” column represents the average accuracy score on the four sub-dataset. tegrated with a Qdrant vector database to hold ex- ternal sources of knowledge retrieved from local corpus, web search, and calculators, designed to answer open-domain clinical questions. Like Al- manac, Lozano et al. (2023) introduced Clinfo.ai, which is an open-source, end-to-end retrieval- augmented LLM (GPT) to answer medical queries using scientific literature summarizations derived from PubMed search engine. We adopt both Al- manac and Clinfo.ai with the same prompt as ours to answer multiple-choice questions to suit the ADQA benchmark. Lastly, we implement a sim- ple retrieval-augmented GPT baseline with CoT prompting similar to our proposed DALK. All the GPT models used are set to GPT-3.5-turbo as de- tailed in the next paragraph, to be consistent. Implementation Details We use the knowledge graph constructed with the generative approach (KGgen) in our main experiment and conduct an ablation study on the knowledge graph with RE method (KGpair) in Section 4.5. We use GPT-3.5- turbo with the version “gpt-3.5-turbo-0301” and set the sampling temperature to 0.7. We utilize 7B versions of all the biomedical LLMs baselines. For RAG methods, we split each document with a max length of 128 and retrieve the top 3 most relevant documents as the support evidence for LLMs to do inference. We set the parameter k in our self- aware knowledge retrieval to 5 and conduct further analysis on it in Section 5.2. 4.3 Main Result Table 2 shows the experimental results on our ADQA benchmark. We note that upon applying our dynamic co-augmentation framework, DALK’s per- formance surpasses that of other biomedical LLMs and RAG methods overall. It consistently achieves either the best or the second-best accuracy score across all sub-datasets and attains the highest AVG score. Furthermore, the substantial improvement over vanilla GPT-3.5-turbo underscores the efficacy of our approach in domain-specific ADQA. Furthermore, we observe that the performance of biomedical-specific LLMs generally lags be- hind that of GPT-3.5-turbo. We attribute this dis- crepancy to the smaller size of these biomedical LLMs. While they may perform adequately in gen- eral medical contexts, they fall short in the AD scenario, which demands more domain-specific knowledge. In the case of GPT-3.5-turbo combined with various RAG methods, it is evident that most RAG methods enhance the models’ performance. Among them, GPT-3.5-turbo with Clinfo.ai yields the most significant improvement, boosting the ac- curacy score from 67.1 to 70.1 compared to vanilla GPT-3.5-turbo. However, it is important to note that the original Clinfo.ai necessitates access to the PubMed API, constituting an external resource. When we disable this access and solely utilize the same corpora as in DALK within the Clinfo.ai re- trieval system, the improvement it brings becomes marginal and incomparable to our method. Due to the space limitation, we put more RAG results with different hyper-parameters in Appendix D. 4.4 Ablation Study on Self-aware Knowledge Retrieval In this section, we evaluate the efficacy of our proposed self-aware knowledge retrieval method through an ablation study. As depicted in Ta- ble 3, we observe that while the dynamic co- augmentation framework without the self-aware
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MedQA MedMCQA MMLU QA4MRE AVG AVG Length 107.4 23.8 342.9 17.6 122.9 GPT-3.5-turbo 50.0 71.9 83.6 62.9 67.1 DALK 57.9 75.2 85.4 71.4 72.6 DALK w/o self-aware knowledge retrieval 56.5 71.0 77.6 77.1 70.6 Table 3: Ablation study results with and without our proposed self-aware knowledge retrieval. knowledge retrieval module still enhances the model’s performance, the overall improvement is less pronounced. Furthermore, we observe that the efficacy of self-aware knowledge retrieval corre- lates with the length of queries within a given con- text. For instance, a notable enhancement in per- formance is evident within the MMLU sub-dataset upon the implementation of self-aware knowledge retrieval. We attribute this to the fact that questions in the MMLU dataset typically contain longer con- texts compared to other medical QA datasets inte- grated into ADQA. Consequently, irrelevant knowl- edge sourced from the context may exacerbate the issue of information noise thus underscoring the ne- cessity for self-aware retrieval. Conversely, within QA4MRE, characterized by shorter query lengths, the application of self-aware knowledge retrieval can even lead to a decline in performance. 4.5 Ablation Study on KG Construction AVG #Triples GPT-3.5-turbo 67.1 - DALK w/ Generative KG 72.6 53,585 DALK w/ RE KG 66.3 171,431 Table 4: Ablation study results with generative construc- tion and RE construction. Table 4 illustrates the results of the ablation study conducted using generatively constructed KG and RE-constructed KG. Surprisingly, despite the RE method yielding a KG with a larger scale and more triples, knowledge sampled from it has unexpect- edly resulted in a non-trivial drop in performance within ADQA. After a manual examination of the two constructed knowledge graphs, we find LLMs with the RE construction method have a strong in- clination to wrongly assign a relationship to two unrelated entities, which has been exposed by the previous studies (Wan et al., 2023). In contrast, the generative construction approach exclusively out- puts triples that LLMs confidently endorse, yield- ing a smaller yet more precise knowledge graph. This trade-off between coverage and accuracy un- derscores the critical importance of denoising in the construction of KGs by LLMs. 5 Further Analysis 5.1 Co-augmentation Analysis Figure 3: The size of the knowledge graph (triplet num- ber) and the KG-augmented GPT-3.5-turbo’s perfor- mance (accuracy) over time. To comprehensively understand how the perfor- mance of LLMs evolves in response to increasing KG sizes, we undertake a detailed co-augmentation analysis. Illustrated in Figure 3, our experiments aim to discern the changing performance trends of LLMs as the knowledge triples accumulate annu- ally. Our findings reveal that our framework effec- tively fosters the co-evolution of LLMs and KG, with the performance of KG-augmented LLMs ex- hibiting a generally upward trajectory as the KG expands. Notably, when we remove the self-aware knowledge retrieval module, this upward trend be- comes less significant. This further implies the importance of sampling and selecting appropriate knowledge for LLMs when the KG’s size increases. 5.2 Hyper-parameter Analysis In this section, we do a hyper-parameter analy- sis on the retrieval number k of our self-aware re- trieval module. We select a group of value for k ([1,3,5,10,20,30]) and present the experiment re- sults in Figure 4. We show the accuracy score on MedQA, MedMCQA, QA4MRE and AVG with different k. We find when k is small, an incre- ment to it can lead to a performance enhancement. After the best performance shows up, continually increasing the value of k will cause a smooth de- crease in the model accuracy score. This result indicates the knowledge ranked in the top positions
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Path-based Sub-graph Answer Baseline - C % DALK -w/o self-aware knowledge retrieval neurofibrillary tangles->FORM BY->microtubule-associated protein tau... ... entorhinal cortex->is a part of->brain->ASSOCIATES->mouse with Alzheimer’s disease->brain region->temporal lobe C % DALK Reranked Triples1: entorhinal cortex ->is a part of ->brain Reranked Triples2: entorhinal cortex ->associates ->mouse with Alzheimer’s disease Reranked Triples3: temporal lobe ->affected by ->Alzheimer’s disease D ! Table 5: A case to show the effectiveness of DALK. The question is: “The area of the brain resistant to Neurofibrillary tangles of Alzheimer’s disease is: A. Visual association areas B. Entorhinal coex C. Temporal lobe D.Lateral geniculate body” Figure 4: Different self-aware knowledge retrieval set- tings in MedQA, MedMCQA, QA4MRE and the aver- age score (AVG). is more helpful while the knowledge ranked behind is something not very useful, thus successfully val- idating the capability of LLMs to do a fine-grained knowledge reranking. Moreover, we find the best k value is correlated with the length of queries in each single dataset. For example, the best performance in MedQA (average query length is 107.4) shows up when k = 10 while the best performance in MedMCQA and QA4MRE shows up when k = 5 and 3 respectively. This is consistent with our findings in Section 4.4 that a longer query corresponds to a larger and noisier sub-knowledge graph. 5.3 Sensitivity Analysis on ADQA Benchmark In this section, we conduct a sensitivity analysis for our constructed ADQA by conducting a leave- one-out evaluation on AD-related keywords. We do it by removing the samples with each keyword in our keyword list and calculating the AVG score of the remaining samples. As the result shown in Table 6, we find not all of the keywords are incorporated in our ADQA benchmark. Notably, Benchmark DALK DALK w/o self-aware knowledge retrieval ADQA 72.6 70.6 w/o “Alzheimer” 72.1 70.4 w/o “Dementia” 72.4 71.3 w/o “APOE” 73.2 71.2 w/o “Amyloid beta” 73.5 70.7 w/o “Aging” 72.9 71.4 w/o “Lipoprotein” 73.1 71.0 w/o “Microglia” 72.8 70.9 Table 6: Sensitivity analysis for ADQA benchmark with a leave-one-out evaluation on AD-related keywords. the keywords “CSF Biomarkers”, “Neurogenesis”, “PET Amyloid”, “PET Tau”, “Tau Phosphorylation” lack corresponding samples in ADQA. We believe one critical work in the future for benchmarking AD-related knowledge is to collect QA samples to cover these missing keywords. Moreover, ana- lyzing the performance variation upon removing samples linked to each keyword offers insight into determining the relevance of the keyword to AD. 5.4 Case Study We put an example in Table 5 to showcase the effi- cacy of DALK. We notice while the path-based sub- graph contains the relevant knowledge to exclude option C, it still involves other irrelevant informa- tion and finally fails to prompt the LLMs to pro- duce the correct answer. In contrast, our self-aware knowledge retrieval method successfully chooses the top 3 most relevant triples for the given problem and results in the correct answer D. 6 Conclusion In this research, we begin by analyzing the main limitations of adopting the existing LLMs-based methods in AD-specific areas. To address these issues, we propose a novel approach in the merging of large language models and knowledge graphs in the context of Alzheimer’s Disease. Our team
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provides an innovative dynamic co-augmentation framework for the refinement of large language models and knowledge graphs. Initially, our ap- proach extracts structural insights from the unstruc- tured scientific literature, crafting a specialized knowledge graph for AD. Subsequently, we employ a coarse-to-fine sampling technique coupled with a unique self-aware knowledge retrieval strategy to pinpoint relevant information from the knowl- edge graph. The extensive evaluation conducted in our constructed ADQA benchmark showcases the effectiveness of our method and provides fur- ther hints into the synergy of LLMs and knowledge graph in the context of AD. In the future, we will do more exploration in adopting and benchmarking LLMs in the AD areas. 7 Limitations In the development of our AD-KG, our primary focus lies in the exploration of two distinct meth- ods for extracting relationships between associated entities. For entity recognition, we employ a strong PubTator annotator directly, without delving into the utilization of LLMs in this context. However, we have observed that LLMs also exhibit promis- ing entity extraction capabilities in Section 3.2.1. We defer the refinement of methods for extract- ing entities for KG construction with LLMs to fu- ture works. Furthermore, a significant contribution of our work is the establishment of the ADQA benchmark. Nonetheless, the datasets utilized in constructing ADQA primarily consist of medical school exam questions, potentially exhibiting a do- main gap from the scientific literature informing AD-KG. One potential remedy is leveraging Pub- medQA (Jin et al., 2019); however, it is hindered by limited data amount. In the future, we will keep gathering AD-related QA samples and expanding the size of our ADQA benchmark. 8 Ethics Statement We have familiarized ourselves with and honour the ethical code set out in the ACL Code of Ethics2. The knowledge graphs constructed in the paper are based on published scientific literature from PubMed. The ADQA dataset used in the study is also derived from publicly available medical QA datasets that are properly cited. We strive to ensure our study upholds ethical principles and not cause any kind of safety or privacy concerns. Although 2https://www.aclweb.org/portal/content/acl-code-ethics not observed in our multiple-choice QA analysis, we recognize the possibility of factual errors and hallucinations when using pre-trained LLMs for medical QA tasks in general, and we do not recom- mend these models be applied in a practical setting at present. References Felix Agbavor and Hualou Liang. 2022. Predicting dementia from spontaneous speech using large lan- guage models. PLOS Digital Health, 1:1–14. Rohan Anil, Andrew M Dai, Orhan Firat, Melvin John- son, Dmitry Lepikhin, Alexandre Passos, Siamak Shakeri, Emanuel Taropa, Paige Bailey, Zhifeng Chen, et al. 2023. Palm 2 technical report. arXiv preprint arXiv:2305.10403. Aparna Balagopalan, Benjamin Eyre, Frank Rudzicz, and Jekaterina Novikova. 2020. To BERT or not to BERT: Comparing Speech and Language-Based Approaches for Alzheimer’s Disease Detection. In Proc. Interspeech 2020, pages 2167–2171. Maciej Besta, Nils Blach, Ales Kubicek, Robert Ger- stenberger, Lukas Gianinazzi, Joanna Gajda, Tomasz Lehmann, Michal Podstawski, Hubert Niewiadomski, Piotr Nyczyk, et al. 2023. Graph of thoughts: Solv- ing elaborate problems with large language models. arXiv preprint arXiv:2308.09687. Zhen Bi, Jing Chen, Yinuo Jiang, Feiyu Xiong, Wei Guo, Huajun Chen, and Ningyu Zhang. 2024. Codekgc: Code language model for generative knowledge graph construction. ACM Transactions on Asian and Low-Resource Language Information Process- ing, 23(3):1–16. Tom Brown, Benjamin Mann, Nick Ryder, Melanie Subbiah, Jared D Kaplan, Prafulla Dhariwal, Arvind Neelakantan, Pranav Shyam, Girish Sastry, Amanda Askell, et al. 2020. Language models are few-shot learners. Advances in neural information processing systems, 33:1877–1901. Salvatore Carta, Alessandro Giuliani, Leonardo Piano, Alessandro Sebastian Podda, Livio Pompianu, and Sandro Gabriele Tiddia. 2023. Iterative zero-shot llm prompting for knowledge graph construction. arXiv preprint arXiv:2307.01128. Jiawei Chen, Hongyu Lin, Xianpei Han, and Le Sun. 2024. Benchmarking large language models in retrieval-augmented generation. In Proceedings of the AAAI Conference on Artificial Intelligence, vol- ume 38, pages 17754–17762. Zeming Chen, Alejandro Hernández Cano, Angelika Romanou, Antoine Bonnet, Kyle Matoba, Francesco Salvi, Matteo Pagliardini, Simin Fan, Andreas Köpf, Amirkeivan Mohtashami, et al. 2023. Meditron-70b: Scaling medical pretraining for large language mod- els. arXiv preprint arXiv:2311.16079.
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A Details of LLMs for KG Table 7 and 8 present examples of our two KG construction methods respectively. For both methods, we adopt a select-or-generate prompt to instruct the LLM whether to choose a relation from hetionet (Him- melstein et al., 2017), a well-built general medical KG, or generate a new one to describe the relationship between two entities. In the RE construction method, we also conduct a type matching (Table 9) for each entity from type name of PubTator to that of Hetionet and ask the LLM to choose from the relation set that corresponds to the two entities’ types (Table 10). Input Read the following abstract, extract the relationships between each entity.You can choose the relation from: (covaries, interacts, regulates, resembles, downregulates, upregulates, associates, binds, treats, palliates), or generate a new predicate to describe the relationship between the two entities. Output all the extract triples in the format of "head | relation | tail". For example: "Alzheimer’s disease | associates | memory deficits" Abstract: Thiamine pyrophosphate (TPP) and the activities of thiamine-dependent enzymes are reduced in Alzheimer’s disease (AD) patients. In this study, we analyzed the relationship between thiamine deficiency (TD) and amyloid precursor protein (APP) processing in both cellular and animal models of TD. In SH-SY5Y neuroblastoma cells overexpressing APP, TD promoted maturation of beta-site APP cleaving enzyme 1 (BACE1) and increased beta-secretase activity which resulted in elevated levels of beta-amyloid (Abeta) as well as beta-secretase cleaved C-terminal fragment (beta-CTF). An inhibitor of beta-secretase efficiently reduced TD-induced up-regulation of Abeta and beta-CTF. Importantly, thiamine supplementation reversed the TD-induced alterations. Furthermore, TD treatment caused a significant accumulation of reactive oxygen species (ROS); antioxidants suppressed ROS production and maturation of BACE1, as well as TD-induced Abeta accumulation. On the other hand, exogenous Abeta(1-40) enhanced TD- induced production of ROS. A study on mice indicated that TD also caused Abeta accumulation in the brain, which was reversed by thiamine supplementation. Taken together, our study suggests that TD could enhance Abeta generation by promoting beta-secretase activity, and the accumulation of Abeta subsequently exacerbated TD-induced oxidative stress.Entity: Thiamine (thiamine), deficiency increases beta- secretase, TPP (Thiamine pyrophosphate), Alzheimer’s disease (AD), patients, amyloid precursor protein (beta-amyloid (Abeta)), neuroblastoma, BACE1 (beta-site APP cleaving enzyme 1), Abeta, ROS (reactive oxygen species), Abeta accumulation, mice. Output: Output Thiamine deficiency | downregulates | TPP, Thiamine deficiency | regulates | Alzheimer’s disease, Alzheimer’s disease | involves | beta-secretase, Amyloid precur- sor protein | produces | beta-amyloid, Amyloid precursor protein | in | neuroblastoma, BACE1 | is | beta-secretase, BACE1 | regulates | beta-amyloid, Abeta | is | beta- amyloid, Abeta | in | mice, Abeta | causes | reactive oxygen species, Abeta | regulates | ROS Table 7: An example to extract knowledge triples using generative construction method.
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Input Read the following abstract, answer the following question. Abstract: Thiamine pyrophosphate (TPP) and the activities of thiamine-dependent enzymes are reduced in Alzheimer’s disease (AD) patients. In this study, we analyzed the relationship between thiamine deficiency (TD) and amyloid precursor protein (APP) process- ing in both cellular and animal models of TD. In SH-SY5Y neuroblastoma cells overexpressing APP, TD promoted maturation of beta-site APP cleaving enzyme 1 (BACE1) and increased beta-secretase activity which resulted in elevated levels of beta-amyloid (Abeta) as well as beta-secretase cleaved C-terminal fragment (beta- CTF). An inhibitor of beta-secretase efficiently reduced TD-induced up-regulation of Abeta and beta-CTF. Importantly, thiamine supplementation reversed the TD- induced alterations. Furthermore, TD treatment caused a significant accumulation of reactive oxygen species (ROS); antioxidants suppressed ROS production and maturation of BACE1, as well as TD-induced Abeta accumulation. On the other hand, exogenous Abeta(1-40) enhanced TD-induced production of ROS. A study on mice indicated that TD also caused Abeta accumulation in the brain, which was reversed by thiamine supplementation. Taken together, our study suggests that TD could enhance Abeta generation by promoting beta-secretase activity, and the accumulation of Abeta subsequently exacerbated TD-induced oxidative stress.Entity: Thiamine (thiamine), deficiency increases beta-secretase, TPP (Thiamine pyrophos- phate), Alzheimer’s disease (AD), patients, amyloid precursor protein (beta-amyloid (Abeta)), neuroblastoma, BACE1 (beta-site APP cleaving enzyme 1), Abeta, ROS (reactive oxygen species), Abeta accumulation, mice. Question: predict the relation- ship between Disease entity "sclerosis" and Disease entity "multiple sclerosis", first choose from the following options: A. resembles B. no-relation C. others, please specify by generating a short predicate in 5 words. Answer: Let’s think step by step: Output 1. Sclerosis is a disease of the central nervous system. 2. Multiple sclerosis is the most common form of sclerosis. 3. So the relationship between sclerosis and multiple sclerosis should be "sclerosis is a kind of multiple sclerosis". So the answer is: A. resembles. So the answer is: A. resembles Table 8: An example to extract the relationship between “sclerosis” and “multiple sclerosis” using RE construction method. PubTator Type Hetionet Type Gene genes Chemical compounds Disease diseases Table 9: Entity type match from PubTator to Hetionet. Type-Type Relations genes-genes ["covaries", "interacts", "regulates"] disease-disease ["resembles"] compounds-compounds ["resembles"] genes-diseases ["downregulates","associates","upregulates"] genes-compounds ["binds", "upregulates", "downregulates"] compounds-diseases ["treats", "palliates"] Table 10: Type-type to relation match in Hetionet.
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B Details of KG for LLMs In this section, we provide detailed input and output for adopting KG to augment LLMs, including path-based and neighbor-based sub-graph sampling results (Table 11), self-aware knowledge retrieval (Table 12), describing sub-graphs with LLMs (Table 13) and inference with sampled knowledge (Table 14). The question we showcase here is “The area of the brain resistant to Neurofibrillary tangles of Alzheimer’s disease is: A. Visual association areas B. Entorhinal coex C. Temporal lobe D.Lateral geniculate body”, which same as the one we use in Section 5.4. Path-based Sub-graph neurofibrillary tangles->FORM BY->microtubule-associated protein tau->BINDS-> (18)F-THK-5117->ADMINISTERED TO->rats->has->Alzheimer’s disease -> Alzheimer’s disease ->affects->human->has->AD->DISEASE OF->Brain entorhinal cortex->is a part of->brain->ASSOCIATES-> mouse with Alzheimer’s disease->brain region->temporal lobe Neighbor-based Sub-graph neurofibrillary tangles->FORM BY->microtubule-associated protein tau Alzheimer’s disease ->causes->neuronal death Alzheimer’s disease ->associates->cognitive decline Alzheimer’s disease ->affects->human Alzheimer’s disease ->has subtype->neurodegenerative diseases Table 11: An example of path-based and neighbor-based sub-graph for the question. Input There is a question and some knowledge graph. The knowledge graphs follow entity->relationship ->entity list format. Graph: neurofibrillary tangles->FORM BY->microtubule-associated protein tau->BINDS-> (18)F-THK-5117->ADMINISTERED TO->rats->has->Alzheimer’s disease ->Alzheimer’s disease -> affects->human->has->AD->DISEASE OF->Brain entorhinal cortex->is a part of->brain->ASSOCIATES-> mouse with Alzheimer’s disease->brain region->temporal lobe} Question: The area of the brain resistant to Neurofibrillary tangles of Alzheimer’s disease is: A. Visual association areas B. Entorhinal coex C. Temporal lobe D.Lateral geniculate body Please rerank the knowledge graph and output at most 5 important and relevant triples for solving the given question. Output the reranked knowledge in the following format: Reranked Triple1: xxx ——>xxx Reranked Triple2: xxx ——>xxx Reranked Triple3: xxx ——>xxx Reranked Triple4: xxx ——>xxx Reranked Triple5: xxx ——>xxx Answer: Output Reranked Triples1: entorhinal cortex ->is a part of ->brain Reranked Triples2: entorhinal cortex ->associates ->mouse with Alzheimer’s disease Reranked Triples3: temporal lobe ->affected by ->Alzheimer’s disease Table 12: An example of our self-aware knowledge retrieval in path-based sub-graph.
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Input There are some knowledge graph paths. They follow entity->relationship->entity format. Reranked Triples1: entorhinal cortex ->is a part of ->brain Reranked Triples2: entorhinal cortex ->associates ->mouse with Alzheimer’s disease Reranked Triples3: temporal lobe ->affected by ->Alzheimer’s disease Use the knowledge graph information. Try to convert them to natural language, respectively. Use single quotation marks for entity name and relation name. And name them as Path-based Evidence 1, Path-based Evidence 2,... Output: Output Path-based Evidence 1: ’Entorhinal cortex’ is a part of ’brain’. Path-based Evidence 2: ’Entorhinal cortex’ associates ’mouse’ with ’Alzheimer’s disease’. Path-based Evidence 3: ’Temporal lobe’ is affected by ’Alzheimer’s disease’. Table 13: An example to describe the self-retrieved path-based sub-graph with the LLM. Input Question: The area of the brain resistant to Neurofibrillary tangles of Alzheimer’s disease is: A.Visual association areas B.Entorhinal coex C.Temporal lobe D.Lateral geniculate body You have some medical knowledge information in the following: ###Path-based Evidence 1: ’Entorhinal cortex’ is a part of ’brain’. Path-based Evidence 2: ’Entorhinal cortex’ associates ’mouse’ with ’Alzheimer’s disease’. Path-based Evidence 3: ’Temporal lobe’ is affected by ’Alzheimer’s disease’. ###Neighbor-based Evidence 1: ’neurofibrillary tangles’ are ’FORMED BY’ ’ microtubule-associated protein tau’. Neighbor-based Evidence 2: ’Alzheimer’s disease’ ’CAUSES’ ’neuronal death’. Neighbor-based Evidence 3: ’Alzheimer’s disease’ ’ASSOCIATES’ with ’cognitive decline’. Neighbor-based Evidence 4: ’Alzheimer’s disease’ ’AFFECTS’ ’human’. Neighbor-based Evidence 5: ’Alzheimer’s disease’ ’HAS SUBTYPE’ ’neurodegenerative diseases’." Answer: Let’s think step by step: Output In Alzheimer’s disease, neurofibrillary tangles are composed of abnormal aggregations of tau protein within neurons, leading to neuronal dysfunction and eventual cell death. The lateral geniculate body is a relay center in the thalamus that processes visual information from the retina to the visual cortex. Since the lateral geniculate body is not directly involved in memory and cognitive function, it is less likely to be affected by neurofibrillary tangles in Alzheimer’s disease compared to other brain regions such as the entorhinal cortex and temporal lobe which play crucial roles in memory and cognitive processing. Therefore, the area of the brain resistant to neurofibrillary tangles of Alzheimer’s disease is the lateral geniculate body (option D) Table 14: An example of inference with selected knowledge C ADQA Benchmark Table 15 shows the detailed prompt of our self-sample method to construct ADQA benchmark. Table 16 gives the statistics of our ADQA benchmark. Input Judge whether the question below is related to Alzheimer’s Disease. Please answer yes or no. Question: Treatable causes of dementia are ___. a).AD b).Hypothyroidism c).Multi-infarct dementia d).SDH e).Hydrocephalus Is the question related to Alzheimer’s Disease? Answer: Output Yes Table 15: An example from MedMCQA to self-sample AD-related QA sample with LLMs.
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Dataset MedQA MedMCQA MMLU QA4MRE Total Number 152 210 49 35 446 Table 16: Statistics of our ADQA benchmark. D Further Experiment for RAG MedQA MedMCQA NMMLU QA4MRE AVG Almanac w/ 256 chunk size 50.0 69.0 67.3 62.9 62.3 Almanac w/ top 10 docuemnt 48.7 68.6 65.3 62.9 61.4 Almanac w/ CoT 50.0 65.7 77.6 65.7 64.7 Clinfo.ai w/ 256 chunk size 48.6 66.7 81.6 65.7 65.7 Clinfo.ai w/ top 5 docuemnt 43.4 68.1 77.6 68.6 64.4 Clinfo.ai w/ CoT 48.7 68.6 79.6 68.6 65.0 Table 17: Further experiment in RAG methods with different hyper-parameter settings.
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Databases and ontologies BioThings Explorer: a query engine for a federated knowledge graph of biomedical APIs Jackson Callaghan 1,†, Colleen H. Xu 1,†, Jiwen Xin1,†, Marco Alvarado Cano1, Anders Riutta 2, Eric Zhou1, Rohan Juneja1, Yao Yao1, Madhumita Narayan1, Kristina Hanspers2, Ayushi Agrawal 2, Alexander R. Pico2, Chunlei Wu 1,*, Andrew I. Su 1,* 1Department of Integrative Structural and Computational Biology, The Scripps Research Institute, La Jolla, CA 92037, United States 2Data Science and Biotechnology, Gladstone Institutes, University of California, San Francisco, CA 94158, United States *Corresponding authors. Department of Integrative Structural and Computational Biology, The Scripps Research Institute, La Jolla, CA 92037, United States. E-mails: cwu@scripps.edu (C.W.) and asu@scripps.edu (A.I.S.) †These authors contributed equally to this work. Associate Editor: Peter N. Robinson Abstract Summary: Knowledge graphs are an increasingly common data structure for representing biomedical information. These knowledge graphs can easily represent heterogeneous types of information, and many algorithms and tools exist for querying and analyzing graphs. Biomedical knowl- edge graphs have been used in a variety of applications, including drug repurposing, identification of drug targets, prediction of drug side effects, and clinical decision support. Typically, knowledge graphs are constructed by centralization and integration of data from multiple disparate sour- ces. Here, we describe BioThings Explorer, an application that can query a virtual, federated knowledge graph derived from the aggregated infor- mation in a network of biomedical web services. BioThings Explorer leverages semantically precise annotations of the inputs and outputs for each resource, and automates the chaining of web service calls to execute multi-step graph queries. Because there is no large, centralized knowledge graph to maintain, BioThings Explorer is distributed as a lightweight application that dynamically retrieves information at query time. Availability and implementation: More information can be found at https://explorer.biothings.io and code is available at https://github.com/bio things/biothings_explorer. 1 Introduction While downloadable files are the most common way to share biomedical data, application programming interfaces (APIs) are another popular and powerful mechanism for data dis- semination. Accessing data through APIs has many comple- mentary advantages relative to downloading local copies of data. APIs typically allow users to query for specific subsets of the data that are of interest. API queries are often highly indexed, leading to efficient data retrieval. Finally, API access allows for easy incorporation of the most up-to-date data into other computational applications or workflows. While APIs offer many advantages in terms of data acces- sibility, these advantages do not immediately translate into efficient data integration. APIs generally follow some com- mon architectures and protocols [e.g. representational state transfer (REST), output in JavaScript Object Notation (JSON)], but alignment at this technical level does not guar- antee either syntactic or semantic interoperability. For ex- ample, APIs can use different identifiers for the same gene, different data structures to represent gene attributes, and different terms to describe the relationships between bio- medical entities. There have been some efforts to define and enforce seman- tic and syntactic standards to achieve data interoperability. Examples of this approach include the Beacon API from the GA4GH consortium (Rambla et al. 2022) and the DAS speci- fication for sharing annotations of genomic features (Dowell et al. 2001). These efforts rely on the active participation of API developers in adopting a community standard for their API endpoints. Here, we explore the use of semantically precise API anno- tations as a complementary approach to achieving API inter- operability. We divided this work into two components. First, we created an extension of the widely used OpenAPI annota- tion standard (http://spec.openapis.org/oas/v3.1.0) to seman- tically describe the APIs’ inputs and outputs, and a registry to organize these API annotations. Second, we created an appli- cation called BioThings Explorer to consume the API meta- data and to execute multi-hop graph queries that span multiple APIs. Together, this architecture allows users to query a large, federated knowledge graph based on an inter- connected network of biomedical APIs. This federated design offers a unique approach for creating knowledge graphs that is complementary to the more common strategy of centraliza- tion and local data integration. Received: 18 April 2023; Revised: 18 August 2023; Editorial Decision: 11 September 2023; Accepted: 12 September 2023 V C The Author(s) 2023. Published by Oxford University Press. This is an Open Access article distributed under the terms of the Creative Commons Attribution License (https://creativecommons.org/licenses/by/4.0/), which permits unrestricted reuse, distribution, and reproduction in any medium, provided the original work is properly cited. Bioinformatics, 2023, 39(9), btad570 https://doi.org/10.1093/bioinformatics/btad570 Advance Access Publication Date: 14 September 2023 Applications Note Downloaded from https://academic.oup.com/bioinformatics/article/39/9/btad570/7273783 by guest on 11 January 2026
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2 A registry of semantically annotated APIs The first step in creating a network of interoperable APIs is to annotate each API in a semantically precise way. We built this API annotation system on the OpenAPI specification, the de facto standard for documenting API metadata in a human- and machine-readable format. OpenAPI describes basic API metadata (e.g. title, description, version, contact info), as well as key information on the operation of the API endpoints (e.g. server URL, endpoint input parameters, endpoint response schemas). However, this basic OpenAPI specification does not include key domain-specific information that is necessary to facilitate downstream API interoperability. Therefore, we defined an OpenAPI extension to capture semantically precise annota- tions of each API endpoint. These annotations include the se- mantic types and identifier namespaces of biomedical entities that are both used in querying (inputs) and found in the re- sponse (outputs), the JSON path to the output identifier val- ues in the JSON response, and the predicate describing the relationship between the input and output entities (Supplementary Fig. S1). We also chose a strategy to map arbitrary JSON data struc- tures to an established biological data model. In this effort, we mapped API output to the Biolink Model (Unni et al. 2022), a community-developed data model that was adopted and extended by the NCATS Translator consortium (Fecho et al. 2022). The Biolink model defines the universe of seman- tic types that can be used in BioThings Explorer, the allowed identifier systems for each semantic type and the allowed predicates. We provided a guide to writing these API annota- tions in our GitHub repository (https://github.com/biothings/ biothings_explorer/blob/main/docs/README-writing-x-bte. md). To annotate and catalog APIs with our OpenAPI extension, we leveraged the SmartAPI registry (https://smart-api.info/) (Zaveri et al. 2017). We created SmartAPI registrations for 34 APIs with semantic annotations. The selection of these APIs that are queried by BioThings Explorer is set in a local, instance-specific configuration file. This collection of API annotations can be thought of as a “meta-knowledge graph” (meta-KG), where the nodes represent types of biomedical en- tities (genes, diseases, drugs) and the edges represent APIs that describe relationships between two types of biomedical enti- ties. The SmartAPI meta-KG currently contains 35 nodes and 1817 edges. (The complete meta-KG is shown in Supplementary Table S1 and a partial rendering is shown in Fig. 1.) 3 API interoperability using BioThings Explorer The second step in creating our federated biomedical knowl- edge graph was to create BioThings Explorer, an engine to au- tonomously query the SmartAPI meta-KG, query the annotated APIs to retrieve associations between biomedical entities, and integrate those APIs’ responses. The input to BioThings Explorer is a query graph and the syntax for encoding the query graph was defined by the NCATS Translator consortium (Fecho et al. 2022). The topology of the query graph and the constraints on its nodes and edges de- fine the query (Fig. 2). BioThings Explorer executes the query in three distinct phases: query-path planning, query-path execution, and inte- gration and scoring. 3.1 Query-path planning For every edge in a query graph, BioThings Explorer consults the SmartAPI registry for APIs that serve those types of associ- ations (Fig. 2). For example, in Fig. 1, associations between diseases and genes can be found using APIs from the Comparative Toxicogenomics Database (Davis et al. 2023) and the Biolink API from the Monarch Initiative (Mungall et al. 2017, Unni et al. 2022), while associations between genes and chemicals can be found using MyChem.Info (https://mychem.info/) (Lelong et al. 2022). The sequence of API calls that can satisfy the original query is a “query-path plan.” 3.2 Query-path execution In this phase, BioThings Explorer programmatically and au- tonomously executes each query in each query-path plan based on the semantic annotations for each API identified in the previous phase. BioThings Explorer calls each API, using the SmartAPI annotation to construct calls with the correct syntax and appropriate input identifier, and maps the API responses to the Biolink Model (Unni et al. 2022). BioThings Explorer also performs ID-to-object translation, which facili- tates the chaining of API calls from one step in the query-path to the next step. This ID translation step is critical when suc- cessive APIs in the query-path plan use different identifiers to represent the same biomedical entity (e.g. NCBI Gene ID versus Ensembl Gene ID). ID translation is currently handled by the Translator Node Normalizer (https://github.com/ NCATSTranslator/Translator-All/wiki/Node-Normalizer). The output of this phase is a set of edges for each step of the query- path, which represent the associations between biomedical enti- ties retrieved from the APIs. 3.3 Integration and scoring In this final phase, these sets of edges from the API queries are assembled into result sub-graphs, each of which matches the topology of the query graph. Each result is then scored based on a variety of factors including number of paths, length of paths, and semantic similarity between concepts based on the Normalized Google Distance (Cilibrasi and Vitanyi 2007). 4 Deployment and usage The BioThings Explorer knowledge graph is entirely com- posed from a federated network of APIs. Because there is no local assembly and storage of a large knowledge graph, BioThings Explorer is a very lightweight application that can be easily deployed on almost any standard personal com- puter. The ability of every user to create a local instance of BioThings Explorer removes centralized bottlenecks associ- ated with large queries and/or heavy usage. The code reposi- tory that describes the installation process is at https://github. com/biothings/biothings_explorer. BioThings Explorer cur- rently relies on two external dependencies—the Node Normalizer service and the Biolink Model (Unni et al. 2022). However, nothing in the BioThings Explorer architecture is reliant on these specific tools, so these dependencies can be substituted for alternatives if desired. For users who prefer not to create a local instance of BioThings Explorer, we also maintain a community instance for general use through the NCATS Translator Consortium (https://explorer.biothings.io/). 2 Callaghan et al. Downloaded from https://academic.oup.com/bioinformatics/article/39/9/btad570/7273783 by guest on 11 January 2026
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5 Discussion Integration of existing data from multiple disparate sources is a key step in assessing the state of current knowledge. There are many existing efforts to create biomedical knowledge graphs by integrating locally downloaded data and standard- izing it using a common data model (Himmelstein et al. 2017; Fecho et al. 2021; Mayers et al. 2022; Wood et al. 2022; Morris et al. 2023). These efforts result in centralized knowl- edge graphs of substantial size, often with millions of nodes and tens of millions of edges. BioThings Explorer offers a unique strategy for data inte- gration, focusing on creating a federated knowledge graph by semantically annotating APIs. Rather than bringing all data into a massive, centralized graph database, this federated de- sign instead allows knowledge to remain behind each resour- ce’s API. Data are retrieved at query time by dynamically executing API calls and semantically parsing the results. This architecture functionally separates data dissemination (through API creation) from data modeling and data integra- tion (through semantic annotations). This approach has several advantages. First, by moving the requirements for interoperability from implementation in code to semantic API annotation, we significantly lower the barrier to participation in our API ecosystem. Second, by sep- arating these roles into distinct layers, we promote the overall modularity of our system. These components can develop and evolve in parallel, and these two roles can even be undertaken by separate teams (e.g. one team semantically annotates an API that was created by another team). Third, this design facilitates an iterative approach to API annotation. Developers and API annotators can first provide a minimal set of API metadata, which can later be extended based on fu- ture needs and use cases. The federated design of BioThings Explorer also has some notable limitations. First, our OpenAPI extensions in SmartAPI to semantically annotate APIs only work on APIs that follow the REST protocol and provide output in JSON Figure 1. A visualization of the meta-KG for BioThings Explorer. The nodes in this graph are the semantic types of biomedical entities that BioThings Explorer can retrieve associations between (limited to the top eight most common semantic types). The edges between nodes show what associations between biomedical entities exist in the semantic API network that is accessible through BioThings Explorer. The edge label shows the number of APIs that can retrieve those types of associations, which is also represented by the edge width. Figure 2. Deconstruction of a query in BioThings Explorer. (A) A free-text representation of a query that can be answered by BioThings Explorer. (B) The graph representation of the same query. The exact syntax of this graph query is specified in the Translator Reasoner API standard described in Fecho et al. (2022) and shown in Supplementary Fig. S2. (C) The deconstruction of the graph query into multiple API calls by consulting the meta-KG in the SmartAPI registry. BioThings Explorer 3 Downloaded from https://academic.oup.com/bioinformatics/article/39/9/btad570/7273783 by guest on 11 January 2026
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format. Second, because the entire federated KG is never in- stantiated in a single place, reasoning and scoring methods that rely on having the entire knowledge graph in memory cannot be used with BioThings Explorer. In sum, we believe that knowledge graphs enable many ex- citing use cases in biomedical research (Nicholson and Greene 2020), and that BioThings Explorer is complementary to existing approaches for assembling knowledge graphs, offer- ing powerful and unique capabilities for both scientific data analysts and tool developers. Supplementary data Supplementary data are available at Bioinformatics online. Conflict of interest None declared. Funding Support for this work was provided by the National Center for Advancing Translational Sciences, National Institutes of Health, through the Biomedical Data Translator program, awards OT2TR003427 and OT2TR003445. Data availability BioThings Explorer is implemented as a NodeJS application. The primary repository for the BioThings Explorer project is at https://github.com/biothings/biothings_explorer, which in turn links to and incorporates other repositories as sub- modules. All code is released under the Apache 2.0 open- source software license. References Cilibrasi RL, Vitanyi PMB. The google similarity distance. IEEE Trans Knowl Data Eng 2007;19:370–83. Davis AP, Wiegers TC, Johnson RJ et al. Comparative toxicogenomics database (CTD): update 2023. Nucleic Acids Res 2023;51: D1257–62. Dowell RD, Jokerst RM, Day A et al. The distributed annotation system. BMC Bioinformatics 2001;2:7. Fecho K, Bizon C, Miller F et al. A biomedical knowledge graph system to propose mechanistic hypotheses for real-world environmental health observations: cohort study and informatics application. JMIR Med Inform 2021;9:e26714. Fecho K, Thessen AE, Baranzini SE et al. Progress toward a universal biomedical data translator. Clin Transl Sci 2022;15:1838–47. Himmelstein DS, Lizee A, Hessler C et al. Systematic integration of biomedical knowledge prioritizes drugs for repurposing. Elife 2017;6:e26726. Lelong S, Zhou X, Afrasiabi C et al. BioThings SDK: a toolkit for build- ing high-performance data APIs in biomedical research. Bioinformatics 2022;38:2077–9. Mayers M, Tu R, Steinecke D et al. Design and application of a knowl- edge network for automatic prioritization of drug mechanisms. Bioinformatics 2022;38:2880–91. Morris JH, Soman K, Akbas RE et al. The scalable precision medicine open knowledge engine (SPOKE): a massive knowledge graph of bio- medical information. Bioinformatics 2023;39(2):btad080. Mungall CJ, McMurry JA, Ko¨hler S et al. The monarch initiative: an in- tegrative data and analytic platform connecting phenotypes to geno- types across species. Nucleic Acids Res 2017;45:D712–22. Nicholson DN, Greene CS. Constructing knowledge graphs and their biomedical applications. Comput Struct Biotechnol J 2020;18: 1414–28. Rambla J, Baudis M, Ariosa R et al. Beacon v2 and Beacon networks: a ‘lingua franca’ for federated data discovery in biomedical genomics, and beyond. Hum Mutat 2022;43:791–9. Unni DR, Moxon SAT, Bada M et al. Biolink Model: a universal schema for knowledge graphs in clinical, biomedical, and translational sci- ence. Clin Transl Sci 2022;15:1848–55. Wood EC, Glen AK, Kvarfordt LG et al. RTX-KG2: a system for build- ing a semantically standardized knowledge graph for translational biomedicine. BMC Bioinformatics 2022;23:400. Zaveri A, Dastgheib S, Wu C et al. smartAPI: towards a more intelligent network of web APIs. In: Blomqvist, E., Maynard, D., Gangemi, A., et al. (eds) The Semantic Web. ESWC 2017. Lecture Notes in Computer Science, 2017, 154–69. https://link.springer.com/chapter/ 10.1007/978-3-319-58451-5_11#chapter-info. 4 Callaghan et al. Downloaded from https://academic.oup.com/bioinformatics/article/39/9/btad570/7273783 by guest on 11 January 2026
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GigaScience , 2025, 14 , 1–12 DOI: 10.1093/gigascience/giae082 RESEARCH Kno wledge gr aph–based thought: a kno wledge graph–enhanced LLM framework for pan-cancer question ans w ering Yichun Feng 1 ,2 ,‡ , Lu Zhou 2 ,‡ , Chao Ma 3 ,‡ , Yikai Zheng 2 , Ruikun He 4 ,5 , * , and Yixue Li 1 ,2 , * 1 Hangzhou Institute for Advanced Study, University of Chinese Academy of Sciences, 310024 Hangzhou, China 2 Guangzhou National Laboratory, Guangzhou International Bio Island, 510005 Guangzhou, China 3 Smartquerier Gene Technology (Shanghai) Co ., Ltd. , 200100 Shanghai, China 4 BYHEALTH Institute of Nutrition & Health, 510663 Guangzhou, China 5 Shanghai Institute of Nutrition and Health, Chinese Academy of Sciences Shanghai, 200030 Shanghai, China ∗Corr espondence addr ess . Ruikun He , BYHEALTH Institute of Nutrition & Health, 510663 Guangzhou, China. E-mail: herk@by-health.com ; Yixue Li, Guangzhou National Laboratory, No. 9 XingDaoHuanBei Road, Guangzhou International Bio Island, 510005 Guangzhou. E-mail: yxli@sibs.ac.cn . ‡ Contributed equally. Abstract Bac kgr ound: In recent years, large language models (LLMs) have shown promise in various domains, notably in biomedical sciences. Howev er, their r eal-world application is often limited by issues like erroneous outputs and hallucinatory responses. Results: We developed the knowledge graph–based thought (KGT) framework, an innov ati v e solution that integrates LLMs with knowl- edge graphs (KGs) to impr ov e their initial r esponses by utilizing v erifia b le information from KGs, thus significantl y r educing factual err ors in r easoning. The KGT fr amew ork demonstr ates str ong adapta bility and performs well acr oss v arious open-source LLMs. No- ta b l y, KGT can facilitate the discov er y of new uses for existing drugs through potential drug–cancer associations and can assist in pr edicting r esistance by anal yzing r elev ant biomarkers and genetic mechanisms. To ev aluate the kno wledge graph question ans wer- ing task within biomedicine , w e utilize a pan-cancer knowledge graph to develop a pan-cancer question answering benchmark, named pan-cancer question answering. Conclusions: The KGT fr amew ork substantiall y impr ov es the accuracy and utility of LLMs in the biomedical field. This study serves as a proof of concept, demonstrating its exceptional performance in biomedical question answering. Ke yw ords: pan-cancer knowledge graph, large language model, knowledge graph question answering, prompt engineering Ke y P oints: r We introduce a framework combining large language models (LLMs) with knowledge gr a phs (KGs) to impr ov e factual accuracy in LLM reasoning. r Our system is a flexible arc hitectur e that seamlessly in- tegr ates v arious LLMs. r Utilizing a pan-cancer knowledge gr a ph, we hav e pr o- posed the first knowledge gr a ph question answering benchmark in the field of biomedicine. r Case studies r e v eal our method enhanced LLMs in ad- dressing biomedical challenges such as drug reposition- ing, r esistance r esearc h, individualized tr eatment, and biomarker analysis. r The method performs favor abl y in comparison to exist- ing methods. Introduction With the increasing prominence of large language models (LLMs) in the field of artificial intelligence, the advent of influential mod- els such as ChatGPT [ 1 ] and Llama [ 2 ] consequently catalyze the de v elopment of a wide array of applications in biomedicine and health care. Ho w ever, LLMs still face the challenge of factual hal- lucination, wher e they gener ate incorr ect statements due to lim- ited inherent knowledge [ 3 ]. Factual hallucination presents a sig- nificant challenge for the practical use of LLMs, especially in real- world scenarios where factual accuracy is crucial. Consequently, there is a growing focus on addressing factual hallucinations in LLMs within the field of natural language processing (NLP) [ 4 , 5 ]. LLMs often struggle to ca ptur e and access factual knowledge, primarily due to 3 aspects: the inability to comprehend ques- tions due to the lack of contextual information, the insufficient knowledge to generate accurate ans wers , and the incapacity to recall specific facts [ 6 ]. Consequently, researchers consider the fine-tuning technique as a solution to address these issues. For example, MedAlpaca [ 7 ] builds upon medical data to fine-tune Stanford Alpaca for applications related to medical question ans wering and dialogue . ChatDoctor [ 8 ] is designed to simulate a conversation between a doctor and a patient by fine-tuning Recei v ed: May 27, 2024. Re vised: A ugust 14, 2024. Accepted: October 2, 2024 © The Author(s) 2025. Published by Oxford Uni v ersity Pr ess GigaScience. This is an Open Access article distributed under the terms of the Cr eati v e Commons Attribution License ( https://cr eati v ecommons.org/licenses/by/4.0/ ), which permits unrestricted reuse, distribution, and reproduction in any medium, provided the original work is pr operl y cited. Downloaded from https://academic.oup.com/gigascience/article/doi/10.1093/gigascience/giae082/7943459 by guest on 11 January 2026
{ "author": "Feng Yichun, Zhou Lu, Ma Chao, Zheng Yikai, He Ruikun, Li Yixue", "creationDate": "D:20250106105103Z", "creationdate": "2025-01-06T10:51:03+00:00", "creator": "OUP", "file_path": "/home/donbr/open-biosciences/biosciences-research/data/raw/giae082.pdf", "format": "PDF 1.4", "keywords": "", "modDate": "D:20260111011531+00'00'", "moddate": "2026-01-11T01:15:31+00:00", "page": 0, "producer": "Acrobat Distiller 24.0 (Windows); modified using iTextSharp.LGPLv2.Core 3.7.4.0", "source": "/home/donbr/open-biosciences/biosciences-research/data/raw/giae082.pdf", "subject": "DOI: 10.1093/gigascience/giae082 GigaScience, 14, 0, 00-00-2025. Abstract: In recent years, large language models (LLMs) have shown promise in various domains, notably in biomedical sciences. However, their real-world application is often limited by issues like erroneous outputs and hallucinatory responses.We developed the knowledge graph&#x2013;based thought (KGT) framework, an innovative solution that integrates LLMs with knowledge graphs (KGs) to improve their initial responses by utilizing verifiable information from KGs, thus significantly reducing factual errors in reasoning. The KGT framework demonstrates strong adaptability and performs well across various open-source LLMs. Notably, KGT can facilitate the discovery of new uses for existing drugs through potential drug&#x2013;cancer associations and can assist in predicting resistance by analyzing relevant biomarkers and genetic mechanisms. To evaluate the knowledge graph question answering task within biomedicine, we utilize a pan-cancer knowledge graph to develop a pan-cancer question answering benchmark, named pan-cancer question answering.The KGT framework substantially improves the accuracy and utility of LLMs in the biomedical field. This study serves as a proof of concept, demonstrating its exceptional performance in biomedical question answering.", "title": "Knowledge graph&#x2013;based thought: a knowledge graph&#x2013;enhanced LLM framework for pan-cancer question answering", "total_pages": 12, "trapped": "" }
2 | GigaScience , 2025, Vol. 14 LLaMA with medical liter atur e. Additionall y, Med-P aLM [ 9 ] shows promising performance on the MedQA exam based on clinical cor por a and human feedback. Meanwhile, aiming at the Chinese medical domain, LLMs such as BenTsao [ 10 ], DoctorGLM [ 11 ], and HuatuoGPT [ 12 ] are developed on the Chinese medical dialogue data. Mor e r ecentl y, Zhongjing [ 13 ] and ChiMed-GPT [ 14 ] adopted full pipeline training from pretraining, SFT, to reinforcement learning with human feedback (RLHF) [ 15 ]. While fine-tuning can reduce hallucinations in LLMs, it brings about considerable train- ing expenses. Additionally, it poses a critical challenge known as catastrophic forgetting. This issue manifests when a model for gets its pr e viousl y learned information as a consequence of parameter modifications during the acquisition of new tasks. This forgetfulness results in a deterioration of performance on prior tasks, consequently constraining the model’s practical applicability [ 16 , 17 ]. In addition to fine-tuning, r esearc hers also enhance the out- put of LLMs through the field of prompt engineering. Prompt en- gineering focuses on the creation and optimization of prompts to impr ov e the effectiv eness of LLMs acr oss v arious a pplications and r esearc h domains [ 18 ]. It can enhance the ca pabilities of LLMs in a wide range of complex tasks, including question answering, sentiment classification, and commonsense reasoning. Chain-of- thought (CoT) prompts [ 19 ] enable complex reasoning capabili- ties by incor por ating intermediate r easoning steps . T he Automatic Pr ompt Engineer (APE) pr oposes an automatic pr ompt gener ation method aimed at enhancing the performance of LLMs [ 20 ]. Prompt engineering offers a straightforw ar d approach to harnessing the potential of LLMs without fine-tuning. On the other hand, knowledge gr a phs (KGs) ar e r epositories of vast quantities of high-quality structured data, offering the po- tential to effectiv el y mitigate the issue of factual hallucinations when integrated with LLMs . Hence , employing KGs for question answering can enhance the precision of the responses and fur- nish a dependable foundation for the factual verification of in- formation produced by LLMs. Knowledge gr a ph question answer- ing (KGQA) has long been a hot r esearc h topic. Befor e the ad- vent of LLMs, certain studies [ 21–23 ] typically begin by r etrie ving a subgr a ph r elated to the question to r educe the searc h space, then perform m ultihop r easoning on this basis . T his r etrie v al- plus-r easoning par adigm has shown its adv anta ges ov er dir ect r easoning acr oss the entir e KG [ 24 , 25 ]. Additionall y, r esearc hers tackle KGQA by parsing the question into a structured query lan- guage (e.g., SPARQL) and using a query engine to obtain accu- rate answers [ 26 , 27 ]. UniKGQA [ 28 ] introduces a unified fine- tuning fr ame work for r etrie v al and r easoning, mor e closel y linking these 2 stages. Ho w ever, traditional KGQA methods usually per- form poorly in accurate semantic understanding and high-quality text generation due to the lack of LLMs for r etrie v al and r eason- ing. Hence, r ecent r esearc h is incr easingl y utilizing external KGs to enhance LLMs in addressing KGQA challenges. For instance, StructGPT [ 29 ] navigates through knowledge graphs by identify- ing pathways from an initial seed entity to the target answer en- tity, while Think-on-Gr a ph (ToG) [ 30 ] intr oduces iter ativ e explo- ration of the knowledge gr a ph, whic h can become inefficient with v ery lar ge KGs. Additionall y, Reasoning on Gr a phs (RoG) [ 31 ] ne- cessitates fine-tuning to accur atel y gener ate and plan the r elation paths. KG-GPT [ 32 ] opts for r etrie ving an entire subgraph from the knowledge gr a ph and then deduces the answer thr ough infer ence. Although these methods hav e ac hie v ed gr atifying r esults in gen- er al ar eas, as shown in Fig. 1 B, when the intermediate entity in the multihop question is unknown, it is impossible to retrieve the a ppr opriate knowledge from the KG. In this article, we intr oduce an innov ativ e fr ame work called knowledge gr a ph–based thought (KGT), whic h integr ates LLMs with KGs through employing LLMs for reasoning on the schema of KGs to mitigate factual hallucinations of LLMs, as shown in Fig. 1 C. Unlike traditional methods, KGT does not dir ectl y r etrie v e factual information based on the question. Instead, it uses LLMs to infer entity information on the schema of the knowledge graph, generating an optimal subgraph based on k e y information di- r ectl y extr acted fr om the question and inferr ed information fr om the sc hema. Subsequentl y, the optimal subgr a ph is used to in- fer the answer to the question through LLMs. KGT r equir es no fine-tuning, offers seamless integration with multiple LLMs, and is plug-and-play, facilitating easy deployment. It demonstrates generalizability, making it adaptable for use with diverse knowl- edge gr a phs . T his fr ame work is tailor ed for wide-r anging a ppli- cations in numerous biomedical challenges, such as (i) enhanc- ing clinical decision-making for physicians and medical organi- zations, (ii) delivering medical advice to patients and health care pro viders , (iii) unco vering crucial biomarkers for early disease de- tection and tailored therapy, and (iv) exploring novel therapeutic applications for existing medications through insights into their mechanisms , side effects , and the biological processes of associ- ated diseases . Furthermore , we utilize the SmartQuerier Oncol- ogy Knowledge Gr a ph (SOKG), a pan-cancer knowledge gr a ph de- veloped by SmartQuerier, to create a benchmark for the KGQA task within biomedicine, named pan-cancer question answering (PcQA). We release this benchmark and its accompanying knowl- edge gr a ph, whic h is a subgr a ph of the SOKG, in [ 33 ]. This bench- mark is curr entl y the sole question-ans wering dataset a vailable in the domain of biomedical knowledge gr a phs. Materials and Methods Kno wledge gr aph introduction In this w ork, w e tac kle the pr oblem of logical r easoning ov er the KG K : E × R that store entities ( E ) and relations ( R ). With- out loss of generality, KG can be organized as a set of triplets { (e 1 , r, e 2 ) } ⊆K, wher e eac h r elation r ∈ R exists between the pair of entities (e 1 , e 2 ) ∈ E × E. We define a relational path { (t 1 , r, t 2 ) } as a sequence of entity types ( T ) and the relation between them, where (t 1 , t 2 ) ∈ T × T . In contrast, a relational chain { (e 1 , r, e 2 ) } refers to a specific set of relational triplets between entities. To further enrich the KG, attribute information is included through pairs (e , at t r ) , where at t r represents an attribute associated with an entity e , thereby enhancing the KG’s semantic richness and precision by incorporating detailed characteristics of each entity. Within the specialized realm of pan-cancer resear ch, w e use a subgr a ph of the SOKG that pr ovides detailed oncological in- formation. As depicted in Table 1 , SOKG includes a collection of over 3 million entities, which is substantially larger than the en- tity count in the compared knowledge graphs, SynLethKG [ 34 ] and SDKG [ 35 ], with 540,012 and 165,062 entities, r espectiv el y. Further- mor e, SOKG’s nearl y 6 million unique concept relations exceed those of SynLethKG and SDKG, which have 2,231,921 and 727,318 r elations, r espectiv el y. Additionall y, SOKG includes 98 distinct at- tribute types, enriching data comprehension and improving the efficiency and precision of queries, a capability not matched by SynLethKG or SDKG, which do not include comparable attributes. For this r esearc h, we utilize only a subgraph of the SOKG, which is available as open data [ 33 ], while the full knowledge graph re- mains proprietary. Downloaded from https://academic.oup.com/gigascience/article/doi/10.1093/gigascience/giae082/7943459 by guest on 11 January 2026
{ "author": "Feng Yichun, Zhou Lu, Ma Chao, Zheng Yikai, He Ruikun, Li Yixue", "creationDate": "D:20250106105103Z", "creationdate": "2025-01-06T10:51:03+00:00", "creator": "OUP", "file_path": "/home/donbr/open-biosciences/biosciences-research/data/raw/giae082.pdf", "format": "PDF 1.4", "keywords": "", "modDate": "D:20260111011531+00'00'", "moddate": "2026-01-11T01:15:31+00:00", "page": 1, "producer": "Acrobat Distiller 24.0 (Windows); modified using iTextSharp.LGPLv2.Core 3.7.4.0", "source": "/home/donbr/open-biosciences/biosciences-research/data/raw/giae082.pdf", "subject": "DOI: 10.1093/gigascience/giae082 GigaScience, 14, 0, 00-00-2025. Abstract: In recent years, large language models (LLMs) have shown promise in various domains, notably in biomedical sciences. However, their real-world application is often limited by issues like erroneous outputs and hallucinatory responses.We developed the knowledge graph&#x2013;based thought (KGT) framework, an innovative solution that integrates LLMs with knowledge graphs (KGs) to improve their initial responses by utilizing verifiable information from KGs, thus significantly reducing factual errors in reasoning. The KGT framework demonstrates strong adaptability and performs well across various open-source LLMs. Notably, KGT can facilitate the discovery of new uses for existing drugs through potential drug&#x2013;cancer associations and can assist in predicting resistance by analyzing relevant biomarkers and genetic mechanisms. To evaluate the knowledge graph question answering task within biomedicine, we utilize a pan-cancer knowledge graph to develop a pan-cancer question answering benchmark, named pan-cancer question answering.The KGT framework substantially improves the accuracy and utility of LLMs in the biomedical field. This study serves as a proof of concept, demonstrating its exceptional performance in biomedical question answering.", "title": "Knowledge graph&#x2013;based thought: a knowledge graph&#x2013;enhanced LLM framework for pan-cancer question answering", "total_pages": 12, "trapped": "" }
Knowledge gr a ph–based thought | 3 A B C Figure 1: Illustr ativ e examples contr asting our work with pr e vious efforts. (A) LLM-onl y-based infer ence , ans wering questions solel y thr ough the inherent knowledge of LLMs. (B) Subgraph-based inference, enhancing LLMs by retrieving the knowledge from KGs based on the question. If intermediate entities are not provided in the multihop question, no appropriate knowledge can be retrieved. (C) Graph schema–based inference, enhancing r etrie v al ca pabilities by r easoning intermediary entity types on the sc hema of the KG, using the knowledge of the KG to enhance LLMs’ responses. Table 1. Comparison of SOKG with SynLethKG and SDKG Entity types Relational types Nodes Edges Attributes SynLethKG 11 24 54,012 2,231,921 0 SDKG 7 12 165,062 727,318 0 SOKG 24 21 3,640,259 10,656,273 98 Task description In order to tackle a div erse arr ay of c hallenges in the field of biomedicine, we have designed 4 categories of problems: 1- hop pr oblems, m ultihop pr oblems, intersection pr oblems, and at- tribute pr oblems, as illustr ated in Table 2 . Based on these 4 types of tasks, we le v er a ge the SOKG to establish a benchmark for the KGQA task within biomedicine, named PcQA. Unlike KGQA tasks in general domains, such as MetaQA [ 36 ] and FACTKG [ 37 ], whic h typicall y pr ovide the entity types of intermediate entities, KGQA problems in the biomedical domain often do not have any information about intermediate entities. Instead, the infor- mation about intermediate entities must be inferred from the question itself rather than being dir ectl y pr ovided, as shown in Supplementary Table S1 . Additionally, our PcQA dataset includes attributes such as whether a drug is tar geted ther a py or if a mutated gene is oncogenic. This makes our tasks slightly more challenging and better suited to the actual needs of biomedical KGQA. One-hop problems One-hop pr oblems involv e single-r elation c hain r easoning, wher e the objective is to deduce the tail entity T ? given a head entity H 1 and a relation R 1 , or to infer the relation R ? when a head en- tity H 1 and a tail entity T 1 are known, as depicted in equations ( 1 ) and ( 2 ). H 1 + R 1 → T ? (1) H 1 + T 1 → R ? (2) Downloaded from https://academic.oup.com/gigascience/article/doi/10.1093/gigascience/giae082/7943459 by guest on 11 January 2026
{ "author": "Feng Yichun, Zhou Lu, Ma Chao, Zheng Yikai, He Ruikun, Li Yixue", "creationDate": "D:20250106105103Z", "creationdate": "2025-01-06T10:51:03+00:00", "creator": "OUP", "file_path": "/home/donbr/open-biosciences/biosciences-research/data/raw/giae082.pdf", "format": "PDF 1.4", "keywords": "", "modDate": "D:20260111011531+00'00'", "moddate": "2026-01-11T01:15:31+00:00", "page": 2, "producer": "Acrobat Distiller 24.0 (Windows); modified using iTextSharp.LGPLv2.Core 3.7.4.0", "source": "/home/donbr/open-biosciences/biosciences-research/data/raw/giae082.pdf", "subject": "DOI: 10.1093/gigascience/giae082 GigaScience, 14, 0, 00-00-2025. Abstract: In recent years, large language models (LLMs) have shown promise in various domains, notably in biomedical sciences. However, their real-world application is often limited by issues like erroneous outputs and hallucinatory responses.We developed the knowledge graph&#x2013;based thought (KGT) framework, an innovative solution that integrates LLMs with knowledge graphs (KGs) to improve their initial responses by utilizing verifiable information from KGs, thus significantly reducing factual errors in reasoning. The KGT framework demonstrates strong adaptability and performs well across various open-source LLMs. Notably, KGT can facilitate the discovery of new uses for existing drugs through potential drug&#x2013;cancer associations and can assist in predicting resistance by analyzing relevant biomarkers and genetic mechanisms. To evaluate the knowledge graph question answering task within biomedicine, we utilize a pan-cancer knowledge graph to develop a pan-cancer question answering benchmark, named pan-cancer question answering.The KGT framework substantially improves the accuracy and utility of LLMs in the biomedical field. This study serves as a proof of concept, demonstrating its exceptional performance in biomedical question answering.", "title": "Knowledge graph&#x2013;based thought: a knowledge graph&#x2013;enhanced LLM framework for pan-cancer question answering", "total_pages": 12, "trapped": "" }
4 | GigaScience , 2025, Vol. 14 Table 2. Four differ ent r easoning types of task. Eac h r easoning type ma y include o v erla pping questions, so the sum acr oss the 4 differ ent reasoning types of the task may exceed the total number of questions Reasoning type Claim example Graph Question number One-hop What types of cancer can be treated with diethylstilbestrol? 243 Multihop What genetic mutations are present in adenoid cystic carcinoma? 124 Intersection Which drugs are ALK in basaloid large cell carcinoma of the lung sensitivity to? 37 Attribute What is the maximum age for recruitment of clinical trials for patients with meningioma? 59 Multihop problems Multihop pr oblems involv e m ultiple-r elation c hain r easoning that can be br oadl y categorized into 2 types . T he first category in volves deducing potential relationships between entities by navigating thr ough indir ect r elations. By examining the indir ect r elations ( R 1 , R 2 ) between a head entity H 1 and a tail entity T 1 , it is possible to infer an unknown or potential relation R ? linking them dir ectl y. This inference process is encapsulated in the following equation: H 1 + T 1 → R 1 + R 2 → R ? (3) The second category extends the reasoning to include the discov- ery of entities themselves b y follo wing a path from a head entity through intermediate relations to a final tail entity. Starting with a head entity H 1 , coupled with an indir ect r elation R 1 , an inter- mediary entity M can be inferred. This intermediary entity M is then applied with an indirect relation R 2 to deduce the final tail entity T ? . This infer ence pr ocess is summarized in the following equation: H 1 + R 1 → M + R 2 → T ? (4) Intersection problems Intersection pr oblems r efer to taking the intersection of multi- ple relational chains. Two head entities ( H 1 , H 2 ) lead to the deduc- tion of 2 types of tail entities ( T 1 , T 2 ) based on different relations ( R 1 , R 2 ). The final tail entity T ? is determined by intersecting these 2 types of tail entities ( T 1 , T 2 ). This inference process is summa- rized as following: H 1 + R 1 → T 1 (5) H 2 + R 2 → T 2 (6) T 1 ∩ T 2 → T ? (7) Attribute problems Attribute problems refer to the attribute information of the en- tity, where the task involves retrieving the attributes of a known head entity H 1 or determining whether the tail entity T 1 , identified through a known head entity H 1 and relation R 1 , satisfies the at- tributes specified in the query, as illustrated in equations ( 8 ) and ( 9 ). H 1 → P H 1 (8) H 1 + R 1 → P T 1 (9) Datasets In the continuously evolving field of biomedical r esearc h, the in- tegration of LLMs with KGs offers a more efficient and effective method for knowledge discovery and utilization, particularly in adv ancing cancer r esearc h. Nonetheless, we note a scarcity of a ppr opriate datasets for e v aluating these sophisticated method- ologies within this field. To address this, we leverage the SOKG to establish a benchmark for the KGQA task within biomedicine, named PcQA. Our questions were carefully crafted by experts based on the content of the knowledge gr a ph. GPT-4 [ 38 ] was then emplo y ed to generate Cypher queries, which were used to re- trie v e answers fr om the knowledge gr a ph. The gener ated Cypher queries and corresponding answers underwent an initial r e vie w by a biomedical PhD candidate, who manually verified and cor- rected the dataset against the knowledge graph. Finally, the en- tire dataset was thoroughly reviewed by 2 biomedical experts to ensure its accuracy and reliability. This multistep process was meticulously designed to uphold the highest standards of qual- ity throughout the dataset creation. This dataset, along with the accompan ying knowledge gr a ph, is completel y open source [ 33 ]. The PcQA includes 405 data entries, covering a wide range of appli- cations in the field of pan-cancer r esearc h, including genetic pre- disposition to cancer, medication tr eatment planning, drug r epo- sitioning, identification of potential drug targets, studies on drug r esistance, and pr edictions of cancer pr ogr ession and metastasis. By deeply exploring cancer-related reasoning and information re- trie v al c hallenges, this dataset can inspir e r esearc hers and clini- cians to gain a deeper understanding of cancer and explore more effectiv e tr eatment methods. KGT fr ame w ork T he o v er all fr ame work of KGT is laid out in Fig. 2 . When users input their question in natural language, the first step is to ana- lyze the question, extracting the main information with the goal of breaking down the question into smaller, more manageable units. This main information is then passed to an LLM, which applies gr a ph r easoning on the sc hema gr a ph of the knowledge gr a ph, yielding the optimal r elational path. Subsequentl y, a r etrie v al statement is generated, and a subgraph is constructed within the Downloaded from https://academic.oup.com/gigascience/article/doi/10.1093/gigascience/giae082/7943459 by guest on 11 January 2026
{ "author": "Feng Yichun, Zhou Lu, Ma Chao, Zheng Yikai, He Ruikun, Li Yixue", "creationDate": "D:20250106105103Z", "creationdate": "2025-01-06T10:51:03+00:00", "creator": "OUP", "file_path": "/home/donbr/open-biosciences/biosciences-research/data/raw/giae082.pdf", "format": "PDF 1.4", "keywords": "", "modDate": "D:20260111011531+00'00'", "moddate": "2026-01-11T01:15:31+00:00", "page": 3, "producer": "Acrobat Distiller 24.0 (Windows); modified using iTextSharp.LGPLv2.Core 3.7.4.0", "source": "/home/donbr/open-biosciences/biosciences-research/data/raw/giae082.pdf", "subject": "DOI: 10.1093/gigascience/giae082 GigaScience, 14, 0, 00-00-2025. Abstract: In recent years, large language models (LLMs) have shown promise in various domains, notably in biomedical sciences. However, their real-world application is often limited by issues like erroneous outputs and hallucinatory responses.We developed the knowledge graph&#x2013;based thought (KGT) framework, an innovative solution that integrates LLMs with knowledge graphs (KGs) to improve their initial responses by utilizing verifiable information from KGs, thus significantly reducing factual errors in reasoning. The KGT framework demonstrates strong adaptability and performs well across various open-source LLMs. Notably, KGT can facilitate the discovery of new uses for existing drugs through potential drug&#x2013;cancer associations and can assist in predicting resistance by analyzing relevant biomarkers and genetic mechanisms. To evaluate the knowledge graph question answering task within biomedicine, we utilize a pan-cancer knowledge graph to develop a pan-cancer question answering benchmark, named pan-cancer question answering.The KGT framework substantially improves the accuracy and utility of LLMs in the biomedical field. This study serves as a proof of concept, demonstrating its exceptional performance in biomedical question answering.", "title": "Knowledge graph&#x2013;based thought: a knowledge graph&#x2013;enhanced LLM framework for pan-cancer question answering", "total_pages": 12, "trapped": "" }
Knowledge gr a ph–based thought | 5 F igure 2: F r ame work of KGT. (A) Question anal ysis. Decompose the question and extr act its k e y information. (B) Gr a ph sc hema–based infer ence. Input the types of the head and tail entities into the gr a ph sc hema of the knowledge gr a ph, complete the gr a ph r easoning, and obtain the optimal r elational path. (C) Subgr a ph construction. Gener ate a query statement and r etrie v e the subgr a ph. (D) Infer ence. Complete the final r easoning and output the results in natural language. Note: The symbol “×” r epr esents content that has been filtered out by the LLM, while “✓ ” denotes the optimal content selected by the LLM. KG thr ough searc h. The r elational c hains and attributes in the subgr a ph ar e then fed back into the LLM to finalize the reason- ing and generate an output in natural language. Question analysis Key information extraction The user inputs a question text ( Q ) in natural language, which is initially deconstructed and parsed. An LLM is applied to ana- lyze the question, resulting in the identification of the head entity name ( H n ), the tail entity type ( T t ), and the attributes of tail entity ( T a ). The prompt for the LLM to extract k e y information from the question is presented in Supplementary Fig. S1 . Retrie ving ke y information from the KG Based on H n , a fixed Cypher format is set to query the head entity type ( H t ), facilitating subsequent reasoning. Graph schema–based inference Construction of a graph based on KG schema Based on the entity types ( E t ) and the relations ( R ) between them in the SOKG, an undirected graph G is established where E t serve as nodes N and R act as edges P. Candidate path sear c h Br eadth-first searc h (BFS) is emplo y ed to identify the shortest paths connecting H t and T t from the constructed graph G. Initi- ate the search at H t , creating a queue to hold nodes encountered along the way. Sim ultaneousl y, form a set to tr ac k nodes that have been visited to avoid r e visiting them. Insert H t into the queue. Con- tinue processing as long as the queue remains nonempty, remov- ing a node from the queue at each step. For each of its unvisited neighbors, enqueue the neighbor, mark it as visited, and log the pathway from H t to this neighbor. Upon arrival at T t , use the accu- mulated path data to compile the set of shortest paths ( SPs ) from H t to T t , with each individual path within the set r eferr ed to as an SP . The nodes in each SP represent entity types, while the edges denote the relationships between these entity types. Optimal path selection By utilizing embedding technology, textual information is mapped into a low-dimensional space, resulting in N-dimensional real- v alue v ectors . T he similarity between each SP and the Q is calcu- lated based on their r espectiv e r eal-v alue v ectors, with the SP ex- hibiting the highest similarity being selected as the optimal path ( OP ). Similarity (Q, SP) = Q · SP ∥ Q∥ × ∥ SP∥ =  n i =1 (Q i × SP i )   n i =1 Q 2 i ×   n i =1 SP 2 i (10) OP = max Q,SP Similarity (Q, SP) (11) Subgraph construction Query statement generation Input H t , H n , T t , T a , and OP into an LLM to generate a query statement, such as Cypher . T ext2Cypher Prompt is presented in Supplementary Fig. S2 . Subgr aph gener ation Enter the query statement in the KG to obtain a reasonable sub- gr a ph. Downloaded from https://academic.oup.com/gigascience/article/doi/10.1093/gigascience/giae082/7943459 by guest on 11 January 2026
{ "author": "Feng Yichun, Zhou Lu, Ma Chao, Zheng Yikai, He Ruikun, Li Yixue", "creationDate": "D:20250106105103Z", "creationdate": "2025-01-06T10:51:03+00:00", "creator": "OUP", "file_path": "/home/donbr/open-biosciences/biosciences-research/data/raw/giae082.pdf", "format": "PDF 1.4", "keywords": "", "modDate": "D:20260111011531+00'00'", "moddate": "2026-01-11T01:15:31+00:00", "page": 4, "producer": "Acrobat Distiller 24.0 (Windows); modified using iTextSharp.LGPLv2.Core 3.7.4.0", "source": "/home/donbr/open-biosciences/biosciences-research/data/raw/giae082.pdf", "subject": "DOI: 10.1093/gigascience/giae082 GigaScience, 14, 0, 00-00-2025. Abstract: In recent years, large language models (LLMs) have shown promise in various domains, notably in biomedical sciences. However, their real-world application is often limited by issues like erroneous outputs and hallucinatory responses.We developed the knowledge graph&#x2013;based thought (KGT) framework, an innovative solution that integrates LLMs with knowledge graphs (KGs) to improve their initial responses by utilizing verifiable information from KGs, thus significantly reducing factual errors in reasoning. The KGT framework demonstrates strong adaptability and performs well across various open-source LLMs. Notably, KGT can facilitate the discovery of new uses for existing drugs through potential drug&#x2013;cancer associations and can assist in predicting resistance by analyzing relevant biomarkers and genetic mechanisms. To evaluate the knowledge graph question answering task within biomedicine, we utilize a pan-cancer knowledge graph to develop a pan-cancer question answering benchmark, named pan-cancer question answering.The KGT framework substantially improves the accuracy and utility of LLMs in the biomedical field. This study serves as a proof of concept, demonstrating its exceptional performance in biomedical question answering.", "title": "Knowledge graph&#x2013;based thought: a knowledge graph&#x2013;enhanced LLM framework for pan-cancer question answering", "total_pages": 12, "trapped": "" }
6 | GigaScience , 2025, Vol. 14 Inference Subgraph inference Based on the relational chains and attribute data in the subgraph, determine the r ele v ance to the question text. Prune an y err oneous information, r etaining onl y the corr ect r elational c hains. Natural language output The LLM divides the subgr a ph into multiple relational chains, each of which outputs a sentence in natural language, and then the LLM generates natural language output. LLMs Inference and Output Prompt is presented in Supplementary Fig. S3 . Results Ev alua tion criteria We use e v aluators based on GPT-4 [ 38 ], BERTScore [ 39 ], and ROUGE [ 40 ] to assess the accuracy of the generated ans wers . As a scoring bot, GPT-4 e v aluates and assigns scores based on the sim- ilarity in meaning between 2 sentences. GPT-4–based Evaluation Pr ompt is pr esented in Supplementary Fig. S4 . BERTScor e e v alu- ates semantic similarity using context-sensiti ve embed dings, of- fering a compr ehensiv e e v aluation of langua ge model outputs. ROUGE, on the other hand, e v aluates the longest common subse- quence (LCS) between the generated text and the reference text, focusing on sequence-based similarity to assess the fluency and the pr eserv ation of semantic content. Baselines To assess the adv anta ges of our fr ame w ork, w e compare it with se v er al a ppr oac hes that can be dir ectl y a pplied for KGQA tasks without fine-tuning. We introduce a straightforw ar d baseline ap- pr oac h, named Base, which is similar to KG-GPT [ 32 ], curr entl y the leading method in the KGQA field, excluding the sentence segmentation step of KG-GPT. Initiall y, this involv es le v er a ging an LLM to r etrie v e r ele v ant information fr om the KG by gener at- ing a query statement. Then, another LLM is used to answer the question with the r etrie v ed information. To enhance the baseline, we incor por ate CoT pr ompting [ 19 ] and in-context learning (ICL) tec hniques [ 41 ], collectiv el y r eferr ed to as CoT&ICL. The pr ompts for these methods are illustrated in Supplementary Table S5 . Ad- ditionally, we implement KG-GPT [ 32 ] to enhance the r etrie v al and r easoning ca pabilities of the LLMs. For a fair comparison, all meth- ods are based on Code-Llama-13B [ 42 ]. To further underscore the efficacy of our fr ame w ork, w e con- duct a compar ativ e anal ysis of KGT, whic h is built upon Code- Llama-13B, a gainst 2 highl y ca pable lar ge langua ge models that ar e pr ominent in the general and biomedical domains: ChatGPT- 3.5 [ 1 ] and Taiyi [ 43 ]. ChatGPT-3.5, a leader in tasks across the general domain, has exhibited competitive performance in a wide r ange of a pplications. To compensate for its limited biomedi- cal kno wledge, w e emplo y 2 methodologies pr e viousl y described, Base and CoT&ICL, as advanced baselines to augment ChatGPT- 3.5’s capabilities. Taiyi, a cutting-edge LLM in biomedicine, pre- trained on 2 trillion tokens, le v er a ges its extensiv e biomedical knowledge base for direct question answering, bypassing the need for knowledge gr a ph r etrie v al. Due to the scarcity of KGQA datasets within the biomedical domain, all experiments are conducted on our ne wl y pr oposed benchmark, named PcQA. Compar a ti v e analysis across different KGQA methods We e v aluated the ca pabilities of v arious methods based on Code-Llama-13B, with the experimental results presented in Ta- ble 3 . The experimental results indicate that the Code-Llama- 13B model, enhanced with KGT, consistently surpasses competing methods across all metrics assessed. Notabl y, KG-GPT impr ov es the F1 score by 15.7% over previous methods CoT&ICL, while our method KGT increases the F1 score by 33% over KG-GPT. Because KG-GPT overlooks the impact of entity types and attributes on an- swers within the biomedical domain, this ac hie v ement positions our a ppr oac h as a pioneering benc hmark in biomedical KGQA, eclipsing pr e viousl y established best pr actices. Compar a ti v e analysis across di v erse LLMs We present a comparative study of KGT applied to Code-Llama- 13B against 2 highly capable LLMs in the general and biomedical domains, with experimental results displayed in Table 4 . Code- Llama-13B, enhanced by KGT, significantly outperforms its peers, ac hie ving the highest marks in e v ery assessment metric: a GPT- 4 Eval score of 92.4, a BERTScore of 97.7, and a ROUGE F1 score of 86.8. Remarkably, our approach’s F1 score surpasses that of ChatGPT-3.5 with the Base method by 52.7%, the CoT&ICL method by 36.3%, and Taiyi’s base model by 67.3%. These results highlight KGT’s substantial contribution to improving the performance of lar ge langua ge models for the pan-cancer KGQA task. Ev en when integrated with open-source general models, KGT exhibits re- markable performance, outstripping both the recognized state- of-the-art closed-source large language models and those specif- icall y tailor ed for the biomedical domain. This showcases KGT’s adeptness at parsing and le v er a ging knowledge gr a ph data, set- ting a new standard for future research and applications in the field. Assessing KGT’s effecti v eness on di v erse LLM platforms To underscore the adaptability and effectiveness of our KGT fr ame work when applied to a range of large language models, we conduct experiments on se v er al LLMs: Zephyr [ 44 ], Llama-2 [ 2 ], and Code-Llama [ 42 ]. The outcomes, illustrated in Fig. 3 , re- veal that while the CoT&ICL techniques significantly boost per- formance in terms of F1 score, our KGT methodology delivers e v en mor e substantial enhancements acr oss all e v aluated mod- els . T his demonstrates not only the effectiveness of CoT&ICL as a performance-enhancing strategy but also highlights the superior advancements and impact of KGT, establishing its dominance and efficiency in knowledge gr a ph question-answering tasks. Ablation study for dissecting the components of KGT In our effort to illuminate the individual contributions of the com- ponents that constitute our KGT fr ame work and their collective impact on enhancing the performance of LLMs, we define 4 foun- dational modules: (i) question analysis for the extraction of piv- otal information, (ii) gr a ph sc hema–based infer ence to identify the optimal relational chains in the knowledge graph, (iii) the generation of query statements to facilitate subgraph construc- tion, and (iv) the inference process coupled with the articulation of results in natural language . T his ablation study, grounded on the Code-Llama-13B model, is meticulously designed to e v alu- ate the efficacy of these components. Since gr a ph sc hema–based infer ence r equir es the pr ocess of question anal ysis, the ques- Downloaded from https://academic.oup.com/gigascience/article/doi/10.1093/gigascience/giae082/7943459 by guest on 11 January 2026
{ "author": "Feng Yichun, Zhou Lu, Ma Chao, Zheng Yikai, He Ruikun, Li Yixue", "creationDate": "D:20250106105103Z", "creationdate": "2025-01-06T10:51:03+00:00", "creator": "OUP", "file_path": "/home/donbr/open-biosciences/biosciences-research/data/raw/giae082.pdf", "format": "PDF 1.4", "keywords": "", "modDate": "D:20260111011531+00'00'", "moddate": "2026-01-11T01:15:31+00:00", "page": 5, "producer": "Acrobat Distiller 24.0 (Windows); modified using iTextSharp.LGPLv2.Core 3.7.4.0", "source": "/home/donbr/open-biosciences/biosciences-research/data/raw/giae082.pdf", "subject": "DOI: 10.1093/gigascience/giae082 GigaScience, 14, 0, 00-00-2025. Abstract: In recent years, large language models (LLMs) have shown promise in various domains, notably in biomedical sciences. However, their real-world application is often limited by issues like erroneous outputs and hallucinatory responses.We developed the knowledge graph&#x2013;based thought (KGT) framework, an innovative solution that integrates LLMs with knowledge graphs (KGs) to improve their initial responses by utilizing verifiable information from KGs, thus significantly reducing factual errors in reasoning. The KGT framework demonstrates strong adaptability and performs well across various open-source LLMs. Notably, KGT can facilitate the discovery of new uses for existing drugs through potential drug&#x2013;cancer associations and can assist in predicting resistance by analyzing relevant biomarkers and genetic mechanisms. To evaluate the knowledge graph question answering task within biomedicine, we utilize a pan-cancer knowledge graph to develop a pan-cancer question answering benchmark, named pan-cancer question answering.The KGT framework substantially improves the accuracy and utility of LLMs in the biomedical field. This study serves as a proof of concept, demonstrating its exceptional performance in biomedical question answering.", "title": "Knowledge graph&#x2013;based thought: a knowledge graph&#x2013;enhanced LLM framework for pan-cancer question answering", "total_pages": 12, "trapped": "" }
Knowledge gr a ph–based thought | 7 Table 3. Comparison of results between KGT and other commonly used methods based on the Code-Llama-13B. The best results are displayed in bold for each indicator ROUGE (%) Method GPT-4 Eval (%) BERTScore (%) Recall Precision F1 score Base 46.6 85.3 25.3 28.5 24.5 CoT&ICL 57.9 88.8 38.9 39.4 37.6 KG-GPT 68.2 93.5 55.2 55.8 53.3 KGT (ours) 92.4 97.7 87.4 87.7 86.8 Table 4. Comparison of KGT based on Code-Llama-13B with results from other commonly used models . T he best results are displayed in bold for each indicator ROUGE (%) Model Method GPT-4 Eval (%) BERTScore (%) Recall Precision F1 score ChatGPT-3.5 Base 65.4 91.0 42.7 32.3 34.1 CoT&ICL 70.3 93.3 57.0 50.6 50.5 Taiyi \ 40.6 85.3 15.4 39.6 19.5 Code-Llama-13B KGT (ours) 92.4 97.7 87.4 87.7 86.8 F igure 3: P erformance of v arious models using differ ent str ategies. tion analysis module cannot be removed in isolation; simulta- neousl y, subgr a ph construction is indispensable for knowledge gr a ph r etrie v al. If the subgr a ph construction module is indepen- dently omitted, the outputs of the initial 2 modules will not impact the final results, making the isolated exclusion of this component illogical. Ther efor e, we intr oduce 3 specific ablated configurations for examination: (i) excluding gr a ph sc hema–based inference (without GSBI), (ii) omitting both question analysis and gr a ph sc hema–based infer ence (without QA&GSBI), and (iii) r e- mo ving question analysis , graph schema–based inference , and subgr a ph construction (without QA&GSBI&SC), effectiv el y by- passing the structured query of the SOKG and relying solely on the LLM’s inherent knowledge for question answering. The results of the ablation study, as shown in Table 5 , demon- strate that when we remove the GSBI, we observe a 20% decrease in the F1 score . Remo ving both GSBI and QA results in an addi- tional 8.6% decrease in the F1 score compared to removing GSBI alone . Furthermore , remo ving GSBI, QA, and SC together leads to a 46% decrease in the F1 score compared to removing just GSBI and QA. The experiments r e v eal that SC is crucial; its absence forces the LLM to r el y solel y on its inher ent knowledge, significantl y r educing effectiv eness. GSBI is also k e y, as it aids in navigating complex multihop questions by providing necessary intermediate entity information for subgr a ph construction. QA is equall y essen- tial, ensuring accurate identification of entities and properties for corr ect subgr a ph construction. All these v ariants under perform Downloaded from https://academic.oup.com/gigascience/article/doi/10.1093/gigascience/giae082/7943459 by guest on 11 January 2026
{ "author": "Feng Yichun, Zhou Lu, Ma Chao, Zheng Yikai, He Ruikun, Li Yixue", "creationDate": "D:20250106105103Z", "creationdate": "2025-01-06T10:51:03+00:00", "creator": "OUP", "file_path": "/home/donbr/open-biosciences/biosciences-research/data/raw/giae082.pdf", "format": "PDF 1.4", "keywords": "", "modDate": "D:20260111011531+00'00'", "moddate": "2026-01-11T01:15:31+00:00", "page": 6, "producer": "Acrobat Distiller 24.0 (Windows); modified using iTextSharp.LGPLv2.Core 3.7.4.0", "source": "/home/donbr/open-biosciences/biosciences-research/data/raw/giae082.pdf", "subject": "DOI: 10.1093/gigascience/giae082 GigaScience, 14, 0, 00-00-2025. Abstract: In recent years, large language models (LLMs) have shown promise in various domains, notably in biomedical sciences. However, their real-world application is often limited by issues like erroneous outputs and hallucinatory responses.We developed the knowledge graph&#x2013;based thought (KGT) framework, an innovative solution that integrates LLMs with knowledge graphs (KGs) to improve their initial responses by utilizing verifiable information from KGs, thus significantly reducing factual errors in reasoning. The KGT framework demonstrates strong adaptability and performs well across various open-source LLMs. Notably, KGT can facilitate the discovery of new uses for existing drugs through potential drug&#x2013;cancer associations and can assist in predicting resistance by analyzing relevant biomarkers and genetic mechanisms. To evaluate the knowledge graph question answering task within biomedicine, we utilize a pan-cancer knowledge graph to develop a pan-cancer question answering benchmark, named pan-cancer question answering.The KGT framework substantially improves the accuracy and utility of LLMs in the biomedical field. This study serves as a proof of concept, demonstrating its exceptional performance in biomedical question answering.", "title": "Knowledge graph&#x2013;based thought: a knowledge graph&#x2013;enhanced LLM framework for pan-cancer question answering", "total_pages": 12, "trapped": "" }
8 | GigaScience , 2025, Vol. 14 Table 5. Ablation study of the KGT fr ame work under Code-Llama-13B ROUGE (%) Method GPT-4 Eval (%) BERTScore (%) Recall Precision F1 score KGT (ours) 92.4 97.7 87.4 87.7 86.8 Without GSBI 71.8 95.5 68.1 69.8 66.8 Without QA&GSBI 69.7 94.7 55.0 66.3 58.2 Without QA&GSBI&SC 24.7 77.4 14.8 12.3 12.2 compared to the complete KGT, indicating that each of the 3 mod- ules is vital for the final performance . Furthermore , such obser- vations confirm that our KGT can indeed le v er a ge knowledge to enhance the final performance of LLMs. Implementation settings Our knowledge gr a ph is quite large, with a complex schema, and typicall y involv es input tokens within 1,300. Our experiment does not r equir e fine-tuning, and the infer ence time is r elated to the model size and computational resources. For example, when us- ing our method, KGT, with the Code-Llama-13B model on an 80 GB A100 GPU, it occupies 33 GB of VRAM. Without any acceleration fr ame works, the infer ence r equir es 4 passes, eac h taking ar ound 20 seconds. Case studies Drug repositioning Drug r epositioning emer ges as a pr omising str ategy to acceler ate the process of drug development. This approach involves identi- fying new therapeutic uses for existing drugs, thereby saving time and r esources typicall y r equir ed for bringing a ne w drug to market [ 45 ]. Our system is capable of investigating the potential reposi- tioning of carteolol for the treatment of hemangiomas . T he exam- ple is shown in Supplementary Table S2 and r elational dia gr am is shown in Fig. 4 A. Utilizing the system’s knowledge gr a ph, a r ela- tional chain is delineated, illustrating that propranolol, another inhibitor of ADRB1, is effectiv el y emplo y ed in the treatment of hemangiomas . T he system harnesses this insight to formulate a hypothesis that carteolol, by virtue of its similar mechanism of inhibition, could be potentially repositioning for treating heman- giomas [ 46 ]. This hypothesis would serve as a precursor to clin- ical trials and r esearc h, potentiall y expediting the availability of an additional ther a peutic option for patients with hemangiomas. Drug resistance resear c h Drug resistance in cancer treatment poses a significant challenge in clinical oncology. Understanding the genetic basis of resistance can lead to mor e effectiv e tr eatment str ategies and personalized medicine a ppr oac hes. Researc h in drug r esistance involv es deter- mining why certain cancer-carrying mutated genes are not re- sponsive to specific drugs and finding ways to overcome this re- sistance [ 47 ]. Our system is capable of exploring drug resistance in cancer. The example is shown in Supplementary Table S3 , and a relational diagram is shown in Fig. 4 B. The KG data indicate that the ALK-p.L1196M m utation, whic h is associated with gas- tric cancer, has a known resistance to nalatinib [ 48 , 49 ]. The LLM processes this information and infers that due to this resistance, nalatinib might not be an effective medication for treating can- cers caused by the ALK-p.L1196M mutation. The case highlights the critical importance of understanding specific gene–drug in- teractions in drug resistance research. It demonstrates how cer- tain gene mutations could render a drug ineffective, which in turn could guide oncologists in choosing alternative treatments or de- v eloping ne w drugs that can bypass or tar get the r esistance mec h- anisms. By accelerating the process of understanding drug resis- tance, these artificial intelligence–driven systems can contribute to impr ov ed patient outcomes and the optimization of cancer tr eatment pr otocols. Individualized treatment Details on individualized tr eatment ar e pr ovided in Supplementary Case Studies A . It is important to note that this example is included solely to illustrate the technical capabilities of the proposed method. The output generated in this example has not been validated for clinical use, and further validation in clinical settings would be r equir ed befor e an y suc h a pplication. Selection and understanding of biomarkers Details on selection and understanding of biomarkers are pro- vided in Supplementary Case Studies B . Discussion In this article, we introduce a novel framework KGT, which em- ploys LLMs for reasoning on the schema of KGs, to enhance the reasoning abilities of LLMs in areas with missing domain data by utilizing domain-specific knowledge gr a phs, suc h as oncology knowledge gr a phs, ther eby addr essing the issue of factual hal- lucinations in LLMs. Our method excels in extr acting, v alidat- ing, and refining factual knowledge throughout the LLMs’ rea- soning process. It seamlessly integrates with various LLMs, in- cluding open-source models like Code-Llama, and enhances the capabilities of LLMs solely through prompt engineering and in- context learning without any fine-tuning. This grants it significant generalizability. We possess an extensive oncology knowledge graph and have established a benchmark based on it to e v aluate the ca pabilities of various methods. When tested on PcQA using various open-source LLMs, the KGT fr ame work performs exceptionall y well, sur passing the current best methods by 33%. This significant improvement positions our a ppr oac h as a pioneering benchmark in biomedical KGQA, setting a new standard that advances beyond previously established best pr actices. Additionall y, thr ough case studies, our a ppr oac h has been shown to effectiv el y pr ovide ther a peutic plans, gener ate v aluable hypotheses for drug r epositioning, identify po- tential drug targets, and study drug resistance . T his underscores the pr actical v alue of the KGT fr ame work in deliv ering insight- ful contributions that aid in the de v elopment and optimization of treatment strategies. Each case study’s conclusions are further v alidated by e vidence fr om pr e viousl y published r esearc h pa pers, enhancing the credibility and impact of our findings. Ho w e v er, it is important to note that the constructed QA dataset and the corresponding published subset of the SOKG Downloaded from https://academic.oup.com/gigascience/article/doi/10.1093/gigascience/giae082/7943459 by guest on 11 January 2026
{ "author": "Feng Yichun, Zhou Lu, Ma Chao, Zheng Yikai, He Ruikun, Li Yixue", "creationDate": "D:20250106105103Z", "creationdate": "2025-01-06T10:51:03+00:00", "creator": "OUP", "file_path": "/home/donbr/open-biosciences/biosciences-research/data/raw/giae082.pdf", "format": "PDF 1.4", "keywords": "", "modDate": "D:20260111011531+00'00'", "moddate": "2026-01-11T01:15:31+00:00", "page": 7, "producer": "Acrobat Distiller 24.0 (Windows); modified using iTextSharp.LGPLv2.Core 3.7.4.0", "source": "/home/donbr/open-biosciences/biosciences-research/data/raw/giae082.pdf", "subject": "DOI: 10.1093/gigascience/giae082 GigaScience, 14, 0, 00-00-2025. Abstract: In recent years, large language models (LLMs) have shown promise in various domains, notably in biomedical sciences. However, their real-world application is often limited by issues like erroneous outputs and hallucinatory responses.We developed the knowledge graph&#x2013;based thought (KGT) framework, an innovative solution that integrates LLMs with knowledge graphs (KGs) to improve their initial responses by utilizing verifiable information from KGs, thus significantly reducing factual errors in reasoning. The KGT framework demonstrates strong adaptability and performs well across various open-source LLMs. Notably, KGT can facilitate the discovery of new uses for existing drugs through potential drug&#x2013;cancer associations and can assist in predicting resistance by analyzing relevant biomarkers and genetic mechanisms. To evaluate the knowledge graph question answering task within biomedicine, we utilize a pan-cancer knowledge graph to develop a pan-cancer question answering benchmark, named pan-cancer question answering.The KGT framework substantially improves the accuracy and utility of LLMs in the biomedical field. This study serves as a proof of concept, demonstrating its exceptional performance in biomedical question answering.", "title": "Knowledge graph&#x2013;based thought: a knowledge graph&#x2013;enhanced LLM framework for pan-cancer question answering", "total_pages": 12, "trapped": "" }
Knowledge gr a ph–based thought | 9 A B C D Figure 4: (A), (B), (C), and (D) r espectiv el y r epr esent the r elational dia gr ams of drug r epositioning, drug r esistance r esearc h, individualized tr eatment, and selection and understanding of biomarkers. wer e specificall y designed to v alidate the effectiv eness of the KGT fr ame work within this study. While the dataset is highly relevant to biomedical applications, its scope is primarily focused on vali- dating the proposed method. T herefore , it ma y not co ver all poten- tial use cases. Additionally, our system curr entl y has the dr awbac k of not performing fuzzy matching; if a drug name is misspelled by e v en 1 letter, it fails to r etrie v e information from the knowledge gr a ph. Ther efor e, we plan to impr ov e this aspect in the future to enhance the system’s usability and reliability. Our ultimate goal is to create a robust framework applicable to the rapidly evolving domain of medical knowledge, supporting health care profession- als in delivering personalized, precise medication tailored to the individual needs of each patient. Finally, we affirm that this study serves as a proof of concept, aiming to showcase the technical feasibility and initial efficacy of the method, which has not been validated in actual clinical prac- tice. In any clinical or medical decision-making, reliance should always be placed on the judgment and guidance of professional health care practitioners. Additional Files Supplementary Table S1. Comparison of PcQA with MetaQA and FACTKG in multihop tasks . T he types of intermediate entities are indicated in bold. Supplementary Table S2. Example of drug repositioning. Supplementary Table S3. Example of drug resistance research. Supplementary Table S4. Example of individualized treatment. Supplementary Table S5. Example of selection and understand- ing of biomarkers. Supplementary Table S6. Prompts for Base and CoT&ICL. Supplementary Fig. S1. Prompt for k e y information extraction. Supplementary Fig. S2. Prompt for query statement generation. Supplementary Fig. S3. Prompt for LLM inference and output. Supplementary Fig. S4. Prompt for GPT-4–based evaluation. Supplementary Fig. S5. (A), (B), (C), and (D) r espectiv el y r epr esent the r elational dia gr ams of drug r epositioning, drug r esistance r e- searc h, individualized tr eatment, and selection and understand- ing of biomarkers. Downloaded from https://academic.oup.com/gigascience/article/doi/10.1093/gigascience/giae082/7943459 by guest on 11 January 2026
{ "author": "Feng Yichun, Zhou Lu, Ma Chao, Zheng Yikai, He Ruikun, Li Yixue", "creationDate": "D:20250106105103Z", "creationdate": "2025-01-06T10:51:03+00:00", "creator": "OUP", "file_path": "/home/donbr/open-biosciences/biosciences-research/data/raw/giae082.pdf", "format": "PDF 1.4", "keywords": "", "modDate": "D:20260111011531+00'00'", "moddate": "2026-01-11T01:15:31+00:00", "page": 8, "producer": "Acrobat Distiller 24.0 (Windows); modified using iTextSharp.LGPLv2.Core 3.7.4.0", "source": "/home/donbr/open-biosciences/biosciences-research/data/raw/giae082.pdf", "subject": "DOI: 10.1093/gigascience/giae082 GigaScience, 14, 0, 00-00-2025. Abstract: In recent years, large language models (LLMs) have shown promise in various domains, notably in biomedical sciences. However, their real-world application is often limited by issues like erroneous outputs and hallucinatory responses.We developed the knowledge graph&#x2013;based thought (KGT) framework, an innovative solution that integrates LLMs with knowledge graphs (KGs) to improve their initial responses by utilizing verifiable information from KGs, thus significantly reducing factual errors in reasoning. The KGT framework demonstrates strong adaptability and performs well across various open-source LLMs. Notably, KGT can facilitate the discovery of new uses for existing drugs through potential drug&#x2013;cancer associations and can assist in predicting resistance by analyzing relevant biomarkers and genetic mechanisms. To evaluate the knowledge graph question answering task within biomedicine, we utilize a pan-cancer knowledge graph to develop a pan-cancer question answering benchmark, named pan-cancer question answering.The KGT framework substantially improves the accuracy and utility of LLMs in the biomedical field. This study serves as a proof of concept, demonstrating its exceptional performance in biomedical question answering.", "title": "Knowledge graph&#x2013;based thought: a knowledge graph&#x2013;enhanced LLM framework for pan-cancer question answering", "total_pages": 12, "trapped": "" }
10 | GigaScience , 2025, Vol. 14 Abbreviations APE: automatic prompt engineer; BFS: breadth-first search; CF: catastr ophic for getting; CoT: c hain of thought; GPT: gener ativ e pr etr ained tr ansformer; ICL: in-context learning; KG: knowledge gr a ph; KGQA: knowledge gr a ph question answering; LLM: large langua ge model; NLP: natur al langua ge pr ocessing; PcQA: pan- cancer question answ ering; RLHF: reinfor cement learning with human feedback; SFT: supervised fine-tuning. Availability of Source Code and Requirements Project name: bioKGQA-KGT r Pr oject homepa ge: https:// github.com/ yichun10/ bioKGQA- KGT.git r Operating system(s): Linux (Ubuntu) r Resource usage in inference ste p: A Lin ux (Ubuntu) system with at least 2 CPU cores and 32 GB of VRAM. The GPU card needs at least 60 GB VRAM (either two 32 GB V100s or one 80 GB A100) r Pr ogr amming langua ge: Shell Script (Bash) with Python 3.10.13 r Other r equir ements: Python 3.10.13 with GPU/CPU support, neo4j 5.13.0 (please see more requirements on GitHub repos- itory) r Licenses: MIT license r Resear ch Resour ce Identifier (#RRID): SCR_025176 Ethical Statement This study involves the generation of a biomedical question- answer dataset derived from a biomedical knowledge gr a ph de- veloped by our team. The knowledge gr a ph has been meticulously constructed using nonpersonalized data obtained from various credible biomedical sources . T he data collection and utilization pr ocesses strictl y compl y with all r ele v ant legal r egulations and ethical guidelines, ensuring the highest standards of data secu- rity and privacy. The dataset adheres rigorously to data protection principles and contains no sensitive personal information or iden- tifiable individual health data. Furthermore, as the data collection and processing activities in this study do not involve human sub- jects, this r esearc h did not r equir e ethical r e vie w or a ppr ov al. Author Contributions Y.F. and L.Z. conceiv ed the pr oject. Y.F. pr oposed a KGQA benc h- mark, de v eloped the KGT fr ame work, implemented the code, con- ducted the experiments, and drafted the manuscript. C.M. con- tributed the SmartQuerier Oncology Knowledge Gr a ph. Y.L. and L.Z. supervised the study. All authors read and a ppr ov ed the final manuscript. Funding This work was supported in part by funds from the Na- tional K ey Researc h and De v elopment Pr ogr am of China (Nos. 2022YFF1202101 and 2023YFC3041600), the Chinese Academy of Sciences Research Fund (No. XDB38050200), and the Self- supporting Pr ogr am of Guangzhou National Labor atory (Nos. SRPG22001 and SRPG22007). Da ta Av ailability We have publicly provided a subset of the SmartQuerier Oncol- ogy Knowledge Gr a ph necessary for r epr oducing the r esearc h. An arc hiv al copy of the code and the subgr a ph of the knowledge gr a ph used in this r esearc h is av ailable via Softwar e Herita ge [ 33 ], and the code and datasets can be accessed via GitHub [ 50 ]. Ad- ditionall y, the pr ompts used in inter actions with LLMs [ 1 , 2 , 38 , 42–44 ] during this r esearc h ar e av ailable in the supplemental ma- terial. For access to the complete SmartQuerier Oncology Knowl- edge Gr a ph data, please contact at service@smartquerier.com. Competing Interests Chao Ma is emplo y ed b y SmartQuerier Gene Technology (Shang- hai) Co., a company active in the biomedical field relevant to the content of this r esearc h. The SmartQuerier Oncology Knowledge Gr a ph (SOKG) used in this study is proprietary to SmartQuerier Gene Technology (Shanghai) Co. The other authors declare that they have no competing interests. References 1. OpenAI. ChatGPT (Nov 30 version) [large language model]. 30 Nov 2022. https:// chat.openai.com/ chat . 2. Touvron H, Martin L, Stone K, et al. Llama 2: Open foundation and fine-tuned chat models [large language model]. 2023. arXiv pre print arXi v:230709288. 19 Jul 2023. https:// doi.org/ 10.48550/a rXiv.2307.09288 . 3. Ji Z, Lee N, Frieske R, et al. Survey of hallucination in natural langua ge gener ation. ACM Comput Surv 2023;55(12):1–38. https: // doi.org/ 10.1145/ 3571730 . 4. Liu T, Zheng X, Chang B, et al. To w ar ds faithfulness in open do- main table-to-text generation from an entity-centric view. In: Yang Qiang, ed. Proceedings of the AAAI Conference on Arti- ficial Intelligence. AAAI Press. Vol. 35; 2021:13415–423. https: // doi.org/ 10.48550/arXiv.2102.08585 . 5. Kang D, Hashimoto T. 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{ "author": "Feng Yichun, Zhou Lu, Ma Chao, Zheng Yikai, He Ruikun, Li Yixue", "creationDate": "D:20250106105103Z", "creationdate": "2025-01-06T10:51:03+00:00", "creator": "OUP", "file_path": "/home/donbr/open-biosciences/biosciences-research/data/raw/giae082.pdf", "format": "PDF 1.4", "keywords": "", "modDate": "D:20260111011531+00'00'", "moddate": "2026-01-11T01:15:31+00:00", "page": 9, "producer": "Acrobat Distiller 24.0 (Windows); modified using iTextSharp.LGPLv2.Core 3.7.4.0", "source": "/home/donbr/open-biosciences/biosciences-research/data/raw/giae082.pdf", "subject": "DOI: 10.1093/gigascience/giae082 GigaScience, 14, 0, 00-00-2025. Abstract: In recent years, large language models (LLMs) have shown promise in various domains, notably in biomedical sciences. However, their real-world application is often limited by issues like erroneous outputs and hallucinatory responses.We developed the knowledge graph&#x2013;based thought (KGT) framework, an innovative solution that integrates LLMs with knowledge graphs (KGs) to improve their initial responses by utilizing verifiable information from KGs, thus significantly reducing factual errors in reasoning. The KGT framework demonstrates strong adaptability and performs well across various open-source LLMs. Notably, KGT can facilitate the discovery of new uses for existing drugs through potential drug&#x2013;cancer associations and can assist in predicting resistance by analyzing relevant biomarkers and genetic mechanisms. To evaluate the knowledge graph question answering task within biomedicine, we utilize a pan-cancer knowledge graph to develop a pan-cancer question answering benchmark, named pan-cancer question answering.The KGT framework substantially improves the accuracy and utility of LLMs in the biomedical field. This study serves as a proof of concept, demonstrating its exceptional performance in biomedical question answering.", "title": "Knowledge graph&#x2013;based thought: a knowledge graph&#x2013;enhanced LLM framework for pan-cancer question answering", "total_pages": 12, "trapped": "" }
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{ "author": "Feng Yichun, Zhou Lu, Ma Chao, Zheng Yikai, He Ruikun, Li Yixue", "creationDate": "D:20250106105103Z", "creationdate": "2025-01-06T10:51:03+00:00", "creator": "OUP", "file_path": "/home/donbr/open-biosciences/biosciences-research/data/raw/giae082.pdf", "format": "PDF 1.4", "keywords": "", "modDate": "D:20260111011531+00'00'", "moddate": "2026-01-11T01:15:31+00:00", "page": 10, "producer": "Acrobat Distiller 24.0 (Windows); modified using iTextSharp.LGPLv2.Core 3.7.4.0", "source": "/home/donbr/open-biosciences/biosciences-research/data/raw/giae082.pdf", "subject": "DOI: 10.1093/gigascience/giae082 GigaScience, 14, 0, 00-00-2025. Abstract: In recent years, large language models (LLMs) have shown promise in various domains, notably in biomedical sciences. However, their real-world application is often limited by issues like erroneous outputs and hallucinatory responses.We developed the knowledge graph&#x2013;based thought (KGT) framework, an innovative solution that integrates LLMs with knowledge graphs (KGs) to improve their initial responses by utilizing verifiable information from KGs, thus significantly reducing factual errors in reasoning. The KGT framework demonstrates strong adaptability and performs well across various open-source LLMs. Notably, KGT can facilitate the discovery of new uses for existing drugs through potential drug&#x2013;cancer associations and can assist in predicting resistance by analyzing relevant biomarkers and genetic mechanisms. To evaluate the knowledge graph question answering task within biomedicine, we utilize a pan-cancer knowledge graph to develop a pan-cancer question answering benchmark, named pan-cancer question answering.The KGT framework substantially improves the accuracy and utility of LLMs in the biomedical field. This study serves as a proof of concept, demonstrating its exceptional performance in biomedical question answering.", "title": "Knowledge graph&#x2013;based thought: a knowledge graph&#x2013;enhanced LLM framework for pan-cancer question answering", "total_pages": 12, "trapped": "" }
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{ "author": "Feng Yichun, Zhou Lu, Ma Chao, Zheng Yikai, He Ruikun, Li Yixue", "creationDate": "D:20250106105103Z", "creationdate": "2025-01-06T10:51:03+00:00", "creator": "OUP", "file_path": "/home/donbr/open-biosciences/biosciences-research/data/raw/giae082.pdf", "format": "PDF 1.4", "keywords": "", "modDate": "D:20260111011531+00'00'", "moddate": "2026-01-11T01:15:31+00:00", "page": 11, "producer": "Acrobat Distiller 24.0 (Windows); modified using iTextSharp.LGPLv2.Core 3.7.4.0", "source": "/home/donbr/open-biosciences/biosciences-research/data/raw/giae082.pdf", "subject": "DOI: 10.1093/gigascience/giae082 GigaScience, 14, 0, 00-00-2025. Abstract: In recent years, large language models (LLMs) have shown promise in various domains, notably in biomedical sciences. However, their real-world application is often limited by issues like erroneous outputs and hallucinatory responses.We developed the knowledge graph&#x2013;based thought (KGT) framework, an innovative solution that integrates LLMs with knowledge graphs (KGs) to improve their initial responses by utilizing verifiable information from KGs, thus significantly reducing factual errors in reasoning. The KGT framework demonstrates strong adaptability and performs well across various open-source LLMs. Notably, KGT can facilitate the discovery of new uses for existing drugs through potential drug&#x2013;cancer associations and can assist in predicting resistance by analyzing relevant biomarkers and genetic mechanisms. To evaluate the knowledge graph question answering task within biomedicine, we utilize a pan-cancer knowledge graph to develop a pan-cancer question answering benchmark, named pan-cancer question answering.The KGT framework substantially improves the accuracy and utility of LLMs in the biomedical field. This study serves as a proof of concept, demonstrating its exceptional performance in biomedical question answering.", "title": "Knowledge graph&#x2013;based thought: a knowledge graph&#x2013;enhanced LLM framework for pan-cancer question answering", "total_pages": 12, "trapped": "" }
An LLM-based Knowledge Synthesis and Scientific Reasoning Framework for Biomedical Discovery Oskar Wysocki1,2, Magdalena Wysocka2, Danilo S. Carvalho2, Alex Bogatu2, Danilo Gusicuma1, Maxime Delmas1, Harriet Unsworth2, André Freitas1,2,3 1Idiap Research Institute, Switzerland 2National Biomarker Centre, CRUK-MI, Univ. of Manchester, United Kingdom 3Department of Computer Science, Univ. of Manchester, United Kingdom Correspondence: firstname.lastname@idiap.ch1firstname.lastname@manchester.ac.uk2 Abstract We present BioLunar, developed using the Lu- nar framework, as a tool for supporting bio- logical analyses, with a particular emphasis on molecular-level evidence enrichment for biomarker discovery in oncology. The platform integrates Large Language Models (LLMs) to facilitate complex scientific reasoning across distributed evidence spaces, enhancing the ca- pability for harmonizing and reasoning over heterogeneous data sources. Demonstrating its utility in cancer research, BioLunar lever- ages modular design, reusable data access and data analysis components, and a low-code user interface, enabling researchers of all program- ming levels to construct LLM-enabled scien- tific workflows. By facilitating automatic sci- entific discovery and inference from heteroge- neous evidence, BioLunar exemplifies the po- tential of the integration between LLMs, spe- cialised databases and biomedical tools to sup- port expert-level knowledge synthesis and dis- covery. 1 Introduction Contemporary biomedical discovery represents a prototypical instance of complex scientific reason- ing, which requires the coordination of controlled in-vivo/in-silico interventions, complex multi-step data analysis pipelines and the interpretation of the results under the light of previous evidence (avail- able in different curated databases and in the litera- ture) (Paananen and Fortino, 2019; Nicholson and Greene, 2020). This intricacy emerges out of the inherent complexity of biological mechanisms un- derlying organism responses, which are defined by a network of multi-scale inter-dependencies (Bog- dan et al., 2021). While more granular data is being generated by the evolution of instruments, assays and methods, and the parallel abundance of experi- mental interventions (Dryden-Palmer et al., 2020), there a practical barrier for integrating and coher- ing this evidence space into a specific context of analysis. Within biomedical discovery, the language in- terpretation capabilities of Large Language Mod- els (LLMs) can provide an integrative framework for harmonising and reasoning over distributed ev- idence spaces and tools, systematising and low- ering the barriers to access and reason over mul- tiple structured databases, textual bases such as PubMed, enriching the background knowledge through specialised ontologies and serving as in- terfaces to external analytical tools (e.g. mechanis- tic/perturbation models, gene enrichment models, etc). In this context, LLMs can serve as a linguis- tic analytical layer which can reduce the syntactic impedance across diverse functional components: once an adapter to an external component is built it can be integrated and reused in different contexts, creating a monotonic increase of functional compo- nents. Complementarily, from a Biomedical-NLP perspective, in order to address real-world prob- lems, LLMs need to be complemented with mech- anisms which can deliver contextual control (e.g. via Retrieval Augmented Generation: RAG: ac- cess the relevant background knowledge and facts) and perform the analytical tasks which are integral to contemporary biomedical inference (’toolform- ing’). Emerging LLM-focused coordination frame- works such as LangChain1, Flowise2 and Lunar3 provide the capabilities to deliver a composition of functional components, some of them under a low-code/no-code use environment, using the ab- straction of workflows. While there are general- purpose coordination frameworks, there is a lack of specialised components for addressing biomedical analyses. In this paper we demonstrate BioLunar, a suite of components developed over the Lunar environment 1https://python.langchain.com 2https://github.com/FlowiseAI/Flowise 3https://lunarbase.ai arXiv:2406.18626v1 [q-bio.QM] 26 Jun 2024
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to support biological analyses. We demonstrate the key functionalities of the platform contextualised within a real-use case in the context of molecular- level evidence enrichment for biomarker discovery in oncology. 2 BioLunar BioLunar enables the creation of LLM-based biomedical scientific workflows using software components with standardised APIs. A workflow is composed of components and subworkflows con- nected through input-output relationships, and are capable of handling multiple inputs. In the user in- terface, components are clustered according to their function (see Fig.1). Creating a workflow does not require programming knowledge since components are predefined and merely require data inputs or parameter settings. However, for users who wish to write custom code, ’Python Coder’ and ’R Coder’ components are provided, enabling the definition of custom methods. These custom components can be saved and subsequently accessed in the ’Custom’ group tab. In the paper we describe an exemplar biomed- ical workflow designed to integrate evidence and infer conclusions from bioinformatics pipeline re- sults. Specifically, the biomedical workflow queries expert knowledge bases (KBs) that continuously compile clinical, experimental, and population ge- netic study outcomes, aligning them with assertions relevant to the significance of the observed gene or variant. It then employs Natural Language Infer- ence (NLI) (via LLM) to integrate and harmonise the evidence space and interpreting the results, cul- minating in a comprehensive summary for the en- tire gene set input. This interpretation takes into account the bioanalytical context supplied by the user. 2.1 Exemplar Workflow Next-generation sequencing (NGS) assays play a pivotal role in the precise characterisation of tu- mours and patients in experimental cancer treat- ments. NGS findings are essential to guide the design of novel biomarkers and cancer treatments. Nevertheless, the clinical elucidation of NGS find- ings subsequent to initial bioinformatics analysis often requires time-consuming manual analysis pro- cedures which are vulnerable to errors. The inter- pretation of molecular signatures that are typically yielded by genome-scale experiments are often supported by pathway-centric approaches through which mechanistic insights can be gained by point- ing at a set of biological processes. Moreover, gene and variant enrichment benefits from heteroge- neous curated data sources which pose challenges to seamless integration. Furthermore, there are different levels of supporting evidence and there- fore prioritising conclusions is crucial. Automating evidence interpretation, knowledge synthesis and leveraging evidence-rich gene set reports are fun- damental for addressing the challenges in precision oncology and the discovery of new biomarkers. 2.2 User interface The user interface facilitates an agile workflow construction by enabling users to select and ar- range components via drag-and-drop from func- tionally grouped categories, such as, i.a.: ’Prompt Query’ featuring NLI components, ’Knowledge Bases’ components, ’Extractors’ for retrieving files from zip archives or extracting text and tables from PDF files, and ’Coders’, which allow for the cre- ation of custom components using Python or R scripts. Components allow for individual execution, edi- tion, or configuration adjustment via a visual inter- face. Workflows can be executed, saved, or shared. Each component has designated input and output capabilities, enabling seamless integration where the output from one can directly feed into another. Users have the flexibility to manually input values if no direct connection is established. Additionally, a component’s output can feed into multiple compo- nents. The system’s architecture supports effortless expansion, adding branches and components with- out affecting the existing workflow, thus facilitating scalable customization to meet changing require- ments. The user interface with an example of a workflow is presented in Fig.1 and in demo video https://youtu.be/Hc6pAA_5Xu8. 2.3 Knowledge bases The current framework integrates a diverse set of knowledge bases which are relevant for pre- cision oncology. To identify gene mutations as biomarkers for cancer diagnosis, prognosis, and drug response, we integrated CIViC4 and On- coKB5. CIViC provides molecular profiles (MPs) of genes, each linked to clinical evidence, with 4https://civicdb.org 5https://www.oncokb.org
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Figure 1: BioLunar interface. An exemplary workflow of Gene Enrichment with an input gene set, knowledge base query and LLM interpretation components. a molecular score indicating evidence quality, as- sessed by human annotators. The Gene Ontology6 (GO) offered gene function insights, and the Hu- man Protein Atlas7 supplied a list of potential drug targets and transcription factors. We employed COSMIC8 for somatic mutation impacts in can- cer, the largest resource in this field. Our analysis also included KEGG9, Reactome10, and WikiPath- ways11 for pathway information, enriching our in- vestigation with scientific literature via PubMed’s API 12. In the following subsections, we showcase ex- amples of components, subworkflows, and work- flows constructed using the BioLunar framework, motivated by the biomarker discovery/precision on- cology themes. 2.4 Construction and reuse of specialised prompts BioLunar employs standard LLM interfaces, al- lowing the use of different models according to users’ preferences. The prompt components allows for the composition of specialised prompt chains which can be later reused, defining a pragmatic pathway for specialised Natural Language Infer- ence (NLI) via prompt decomposition/composition. This approach allows for the creation of reasoning 6https://geneontology.org 7https://www.proteinatlas.org 8https://cancer.sanger.ac.uk/cosmic 9https://www.kegg.jp/kegg/ 10https://reactome.org 11https://www.wikipathways.org 12https://pubmed.ncbi.nlm.nih.gov chains that combines user’s instructions with the results of database queries and analyses from spe- cialised tools within the context of the study. An instantiated example of the Azure Open AI prompt is described in Fig.1. 2.5 Subworkflow component The subworkflow component enables the reuse of an existing workflow within another workflow, functioning as a component with specified inputs and outputs. This feature simplifies the composi- tion of more complex workflows and avoids the repetition of defining identical steps for the same task. Subworkflows can be selected like other com- ponents from the left panel in the interface, offering access to all available workflows for easy integra- tion. Examples of subworkflows are presented in Fig.2,3. 2.6 Gene Enrichment subworkflow One example of a specialised subworkflow is the Gene Enrichment subworkflow (Fig.1,2A) begins with uploading the targeted gene sets. Then a component accesses a specific KB — such as Gene Ontology, KEGG, Reactome, or WikiPath- ways—defined by the user, using gprofiler API13. This component identifies gene groups with a sta- tistically significant overlap with the input gene set, according to a Fisher’s test, and calculates p-values, recall, and precision. The user then specifies a vari- able to rank these groups and selects the top N for further analysis. The output includes both a inter- 13https://biit.cs.ut.ee/gprofiler/page/apis
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Gene Enrichment Sort and select top N Upload set of genes Interpret results (LLM) Save results Human Protein Atlas Compute overlap and statistics Upload set of genes Interpret results (LLM) Save results Query Human Protein Atlas Run Gene Enrichment Provide 'Context' Provide 'Context' A) B) Figure 2: A) Gene Enrichment workflow - uses the gprofiler API to access i.a. Gene Ontology, KEGG, WikiPathways, Reactome; B) Human Protein Atlas workflow. Compares and interprets the input and reference gene sets. pretation performed by an NLI component (through LLM) and a table featuring the names, descriptions, and statistics of the top N selected groups. 2.7 Human Protein Atlas subworkflow In the Human Protein Atlas subworkflow, given a gene set, an associated external KB is queried by selecting ‘Transcription factors’ from the HPA database using a dedicated query-database connec- tor. A reusable component, ’Analyze overlap’, then identifies genes that overlap and calculates relevant statistics. Similarly to the Gene Enrichment sub- workflow, the results are interpreted by an prompt- based NLI component and presented alongside a table summarising the findings (Fig.2B,A.7). 2.8 CIVIC subworkflow This subworkflow exemplifies a more complex com- position of components (Fig.3). This subworkflow initiates by querying the CIVIC database for input genes, yielding, among other things, gene descrip- tions in clinical contexts, and their variants and molecular profiles (MPs), which are essential for the final interpretation. Additionally, users spec- ify the analysis context, including aspects such as cancer types or subtypes, treatments, populations, etc. Initially, gene descriptions are analysed by a prompt-based NLI component within this defined context. Subsequently, MPs scored below a prede- fined threshold (set at a MP score of 10) are tagged as less known, reflecting lower scientific evidence and ranking by CIVIC annotators. The evidence supporting these lesser-known MPs is then inter- preted by a prompt-based NLI component, con- sidering the broader analysis context. Conversely, evidence from well-known MPs, scoring above 10, undergoes a similar interpretation process. For genes without identified MPs in CIVIC, a sequence of components perform further evidence retrieval from PubMed. An NLI module gener- ates context-based keywords for PubMed queries, which are combined with the names of genes lack- ing MPs. A ’PubMed search’ component then re- trieves N publications, including metadata, cita- tion counts and MeSH terms (used later for context alignment validation). The abstracts of these pub- lications are interpreted by an NLI module in the context of the analysis. All clinical evidence interpretations are then suc- cinctly summarised by via a prompt component, taking into account the context of the analysis. These interpretations, along with tabular results, constitute the output. 2.9 Bioworkflow - comprehensive analysis for a set of genes. The exemplar bioworkflow composes multiple sub- workflows (Fig.4), each dedicated to a specific multi-step and specialised task, which are typically defined by the composition of heterogeneous com- ponents, most commonly connectors and query in- stance components to specialised databases (e.g.
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Workflow - CivicDB analysis Discovery / new knowledge Get genes  details and molecular profiles Upload set of genes Save results Provide 'Context' Interpret genes in the context (LLM) Identify well known molecular profiles Identify molecular profiles without evidence Query PubMed and select top N Prepare PubMed subquery (LLM) Interpret publications in the context (LLM) Interpret all evidence in the context (LLM) Identify less known molecular profiles Interpret all evidence in the context (LLM) Summarize all interpretations in the context (LLM) Query CIVIC database Figure 3: CIVIC evidence analysis workflow. prompt-based NLI components are fed by both the results and context of the analysis in order to produce relevant evidence-based conclusions. CIVIC, HPA, PubMed, OncoKB), external spe- cialised analytical tools (toolformers for gene en- richment analysis) and chains of specialised in- terpretation prompts (e.g. selection, filtering, ex- traction, summarisation). This setup forms a com- prehensive workflow which exemplifies the close dialogue between LLMs and genomic analysis, en- compassing gene enrichment, comparison with ref- erence gene sets, and access to evidence within an experimental medicine setting. Additionally, it queries PubMed publications within the CIVIC component to seek evidence for molecular profiles not yet described. Its componentised architecture facilitates the extensibility of the workflow with new sources, prompts and external tools. Conclu- sions drawn from each subworkflow are interpreted within the analysis context, being integrated in a comprehensive summary. All findings are com- piled in a report, exported as a PDF file. 2.10 Software description BioLunar uses the LunarVerse backend for its op- erations. LunarVerse is downloaded and installed by the setup script included with the demonstration code. Some of its components need user specific configuration to work, such as private API keys, which are defined in a configuration file indicated in the setup instructions. LunarVerse is distributed un- der a open software license. The workflow can also be operated via a graphical interface (LunarFlow) Running a workflow can be done in two ways: i) directly, by calling the LunarVerse engine on a specified workflow descriptor file; ii) through the Web interface, by pressing the “Run” button. The first way is the default one in the demonstra- tion code. It returns a copy of the workflow descrip- tor, with all component output fields filled, which is then used to extract and filter the desired outputs, based on the component labels. It is also the best way to automate multiple workflow runs and to integrate their outputs into other systems.The sup- porting code is available at https://github.com/ neuro-symbolic-ai/lunar-bioverse-demo. 2.11 Report The Bioworkflow, as outlined in point 2.9, gener- ates a report in PDF (Fig.5) format that begins by outlining the context of the study, analysis details, dates, and software versions at the top. The report is enhanced with hyperlinks for easy navigation to specific sections. A "General Statistics" table provides a compre- hensive overview of key metrics aggregated from all components, aiming to consolidate information for each gene throughout the analysis, with hyper- links directing to the report sections where this information originates. Subsequent sections categorise genes into var-
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Provide 'Context' CIVIC subworkflow Conclusion (LLM) Results (table) Report Overall conclusion  (LLM) HPA subworkflow Conclusion (LLM) Results (table) COSMIC subworkflow Conclusion (LLM) Results (table) OncoKB subworkflow Conclusion (LLM) Results (table) Conclusion (LLM) Gene Enrichment        subworkflow Conclusion (LLM) Results (table) Conclusion (LLM) Results (table) Conclusion (LLM) Results (table) Results (table) Upload set of genes GE summary (LLM) Nik-Zainal-93 subworkflow Conclusion (LLM) Results (table) Figure 4: Diagram of the Bioworkflow. ious tables based on biological aspects and the KBs consulted. These include Molecular Function for genes sharing ontologies, drug target checks based on the Human Protein Atlas, assessments of cancer-related genes, Pathway Analysis and Mapping via WikiPathways, and classification of gene alterations by clinical relevance. By correlat- ing genes with known functional information, the workflow identifies statistically significant enriched terms and summarizes these findings using LLM, which also furnishes evidence. LLM interprets each table, offering textual con- clusions relevant to the analysis context. A final summary, crafted using LLM, synthesizes all re- sults within the given context. Importantly, all LLM interpretations are grounded in concrete ev- idence, with sources cited alongside the narrative. This approach underscores the rigor of the analysis by highlighting distinct sources that substantiate the relevance of each gene and variant. 3 Case study To demonstrate the capabilities of the Biowork- flow, we analyzed outputs in two different scenar- ios, each producing a distinct set of genes from separate bioinformatics analyses. We entered these gene sets along with their analysis contexts into the Bioworkflow and executed it. Subsequently, we qualitatively assessed the output reports (see Fig.A.8,A.9), considering both the statistical data and the interpretations provided by the prompt- based NLI modules. In Scenario 1, the user aims to explore the unique molecular characteristics of HER2-low breast can- cer to determine if it constitutes a distinct category within breast cancer types, where the input genes are ERBB2, ESR1, PIK3CA, CBFB, SF3B. The report shows genomic alterations and genomic sig- natures that were identified, including ERBB2 am- plification, mutations in PIK3CA and ESR1, which are important biomarkers in the selection of breast cancer treatment. For the remaining two genes, evidence was found confirming that these are new, significantly mutated genes for which there is pre- clinical evidence of actionability in clinical prac- tice. In Scenario 2, the user aims to discover new genes that could lead to more accurate breast can- cer diagnoses, enhancing treatment strategies and addressing the disease’s complexity. His numeri- cal analysis resulted in a set of genes (DIXDC1, DUSP6, PDK4, CXCL12, IRF7, ITGA7, NEK2, NR3C1) that require investigation. The report in- forms that none of the genes is an oncogene (con- firmation according to OncoKB), two of the genes
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Cancer type:                Breast Cancer   Sample size: 27   Cancer type: breast cancer   Biopsy site: primary site   Center:   Informed consent:   Sample sent: Export report Analysis run date: 2024-02-24 Pipeline version: v2.0. details   Context:   Breast cancer (BC) presents a significant global health challenge, with its incidence steadily rising and mortality rates remaining high. Its heterogeneous nature complicates treatment strategies, contributing to issues like recurrence and drug resistance. more Navigation Menu Gene set dashboard Context Toolbox Individual gene description ERBB2 Gene Gene Info Oncogene, more Variant ERBB2 L755S Functional relevance evidence Evidence (curated): >Oncogenic, OncoKB >Sensitivity/Response, Resistance, CIViC Hallmark Yes, more ERBB2 D769Y Evidence (curated): >Oncogenic, OncoKB >Sensitivity/Response, CIViC ERBB2 R143G Others/Inconclusive: >Inconclusive/weaker evidence, OncoKB HPA: drug targets FDA approved drug target, more Evidence supporting the variant functional relevance Current biomarker clinical relevance Cancer biomarkers reported at present Investigational, 1 assertions Cancer repurposing, 4 assertions Hypothetical, 2 assertions Investigational, 1 assertions Hypothetical, 1 assertions General Statistics General Statistics ERBB2 This gene is considered an oncogene. ERBB2, a receptor tyrosine kinase, is altered by mutation, amplification and/or overexpression in various cancer types, most frequently in breast, esophagogastric and endometrial cancers. more gene info Gene mutations based on previous pan-cancer cohorts ERBB2 L755S http://oncokb.org/#/gene/ERBB2/alteration/L755S Effect: Oncogenic ERBB2 L755S Please check the original assertions provided by each knowledgebase listed below http://oncokb.org/#/gene/ERBB2/alteration/Oncogenic Mutations Biomarker: ERBB2 Oncogenic Mutations Effect: drug Responsive Evidence level: LEVEL_3A Drug: Neratinib Disease: Breast Cancer Gene with the hallmark description This gene has a hallmark. more This gene has a stimulating effect via individual cancer hallmarks. Interactive pop-up windows Access to external resources with the original evidence Gene-detailed view 1 1 3 3 2 2 4 4 Final conclusion generated by LLM ERBB2, commonly referred to as HER2, is amplified and/or overexpressed in 20-30% of invasive breast carcinomas. HER2-positive breast cancer is treated in a separate manner from other subtypes of breast cancer and commonly presents as more aggressive disease. Metastatic HER2-positive breast cancer is now commonly treated with HER2-targeted therapy. Apart from being amplified/overexpressed, ERBB2 activating mutations have been shown to have clinical importance in HER2-negative breast cancer. These mutations have shown sensitivity to the tyrosine kinase inhibitor neratinib, and highlight the importance of clinical sequencing efforts in treating breast cancer. ERBB2 L755S was one of the first ERBB2 variants to be functionally classified (Bose et al. 2013). This mutation was not shown to be an activating mutation, unlike many of the other variants queried. This mutation was also shown to confer resistance to the tyrosine kinase inhibitor lapatinib in MCF10A cell lines. Final LLM Conclusion LLM Description ERBB2 (Erb-B2 Receptor Tyrosine Kinase 2) is a Protein Coding gene. Among its related pathways are Drug-mediated inhibition of ERBB2 signaling and Signaling by ERBB2 KD Mutants. Gene Ontology (GO) annotations related to this gene more LLM Description Figure 5: The BioLunar report’s overview, produced by Bioworkflow. are potential drug targets and one is FDA approved drug targets. According to the KEGG-based enrich- ment analysis, these genes were mainly enriched through several signaling pathways including tu- mor necrosis factor (TNF) signaling pathway. Us- ing LLMs in conjunction with a PubMed search component, papers were searched in PubMed that describe various gene variants and the genes have been indicated as prospective biomarkers associ- ated with breast cancer. Note that in scenario 2, for genes lacking molec- ular profiles in the KB, a search in PubMed was conducted. This approach enables the workflow to automatically uncover and search for non-obvious and previously unknown relationships. Essentially, if a gene is absent from the database, it suggests that its relevance is relatively novel and not yet documented. Therefore, seeking out the most re- cent publications that describe this gene within the analysis context represents a significant advantage, provided by the workflow that integrates various components. 4 Related Work Bioinformatics Pipelines Over the past decade, three scientific workflow management systems such as Galaxy (gal, 2022), Snakemake (Köster and Rahmann, 2012), and Nextflow (Di Tommaso et al., 2017), have been instrumental to bioinfor- maticians to systematise their complex analytical processes. Nextflow targets bioinformaticians and facilitates gene enrichment analysis, annotate bi-
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ological sequences, and perform gene expression analysis by including modules supported by various bioinformatics tools. These workflow systems are currently centred around the composition of spe- cialised bioinformatics software, configuration pa- rameters and supporting datasets, facilitating reuse and reproducibility. In contrast, this paper explores the concept on using LLMs within a specialised workflow environment to support the interpretation and integration of multiple analytical processes. 5 Conclusion In this paper we provided a demonstration of a scientific workflow based on LLMs to support spe- cialised gene analyses using oncology and gene enrichment as a driving motivational scenario. The framework is built using the Lunar framework and allows for the composition of specialised an- alytical workflows, integrating external databases (Retrieval Augmented Generation), external tools (ToolFormers) and contextualised chains of LLM- based interpretation. The paper highlights that a workflow environment with specialised compo- nents for RAG, ToolFormers and a set of spe- cialised prompts-based Natural Language Infer- ence can serve as the foundation for streamlining and automating complex analytical process within a biomedical setting. . We showcase analytical applications within the biomedical domain, partic- ularly in oncology, constructively progressing to- wards more complex gene analysis workflows. The developed bioworkflow demonstrates the LLMs can be instrumental in enabling a complex end- to-end highly-specialised analytical workflow, in a reproducible manner, supporting the integration of heterogeneous evidence, synthesising conclu- sions and while simultaneously documenting and linking to the data sources within a comprehensive output report. The proposed workflow is based on a low-code paradigm that enables domain experts, regardless of their programming skills, to construct and scientific workflows enabled by generaqtive AI amethods. Limitations • The current demonstration uses external LLM- based APIs but can be adapted to open source LLM models. • The LLM-based inferences require a critical supporting quantitative evaluation and halluci- nations are possible. The current workflow is motivated by a hypothesis generation process, which is fully human supervised and does not have direct clinical applications. Acknowledgements This work was partially funded by The Ark foundation, by the European Union’s Horizon 2020 research and innovation program (grant no. 965397) through the Cancer Core Europe DART project, and by the Manchester Experimental Can- cer Medicine Centre and the NIHR Manchester Biomedical Research Centre. References 2022. The galaxy platform for accessible, reproducible and collaborative biomedical analyses: 2022 update. Nucleic Acids Research, 50(W1):W345–W351. Paul Bogdan, Gustavo Caetano-Anollés, Anna Jolles, Hyunju Kim, James Morris, Cheryl A Murphy, Catherine Royer, Edward H Snell, Adam Steinbren- ner, and Nicholas Strausfeld. 2021. Biological Net- works across Scales—The Theoretical and Empirical Foundations for Time-Varying Complex Networks that Connect Structure and Function across Levels of Biological Organization. Integrative and Compara- tive Biology, 61(6):1991–2010. Paolo Di Tommaso, Maria Chatzou, Evan W Floden, Pablo Prieto Barja, Emilio Palumbo, and Cedric Notredame. 2017. Nextflow enables reproducible computational workflows. Nature biotechnology, 35(4):316–319. K.D. Dryden-Palmer, C.S. Parshuram, and W.B. Berta. 2020. Context, complexity and process in the im- plementation of evidence-based innovation: a realist informed review. BMC Health Services Research, 20(81):1472–6963. Johannes Köster and Sven Rahmann. 2012. Snake- make—a scalable bioinformatics workflow engine. Bioinformatics, 28(19):2520–2522. David N. Nicholson and Casey S. Greene. 2020. Con- structing knowledge graphs and their biomedical ap- plications. Computational and Structural Biotechnol- ogy Journal, 18:1414–1428. Jussi Paananen and Vittorio Fortino. 2019. An omics perspective on drug target discovery platforms. Brief- ings in Bioinformatics, 21(6):1937–1953.
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A Appendix
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Scenario 1 Context: The analysis focuses on HER2-low breast cancer (HLBC), a subtype that challenges traditional classifications based on HER2 expression and ERBB2 amplification. Despite being operationally defined, HLBCs constitute a significant portion of breast cancers, particularly among estrogen receptor- positive tumors. This study aims to elucidate the molecular characteristics of HLBCs, examining their mutational and transcriptional profiles. The research also investigates potential heterogeneity within HLBCs and compares their genomic landscape with HER2-positive and HER2-negative breast cancers. By providing insights into the distinct molecular features of HLBCs, this analysis seeks to establish whether they represent a unique entity in breast cancer pathology. List of genes: ERBB2, ESR1, PIK3CA, CBFB, SF3B1 Scenario 2 Context: Breast cancer (BC) presents a significant global health challenge, with its incidence steadily rising and mortality rates remaining high. Its heterogeneous nature complicates treatment strategies, contributing to issues like recurrence and drug resistance. Biomarkers play a crucial role in diagnosing and managing BC, aiding in personalized treatment approaches. However, existing biomarkers have limitations, necessitating the exploration of novel markers, particularly in the realm of molecular and genetic analysis. This study focuses on identifying genes with potential diagnostic utility in breast cancer, aiming to contribute to the development of more effective biomarkers and therapies, including immunotherapies, to combat this disease. List of genes: DIXDC1, DUSP6, PDK4, CXCL12, IRF7, ITGA7, NEK2, NR3C1 Figure A.6: User-defined context of the analysis, including aspects like cancer types or subtypes, treatments, populations, for Scenario 1 and 2. Figure A.7: Human Protein Atlas workflow in the BioLunar interface.
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Cancer type:                Breast Cancer   Sample size: 5   Cancer type: breast cancer   Biopsy site: primary site   Center:   Informed consent:   Sample sent: Export report Analysis run date: 2024-02-24 Pipeline version: v2.0. details   Context:   The analysis focuses on HER2-low breast cancer (HLBC), a subtype that challenges traditional classifications based on HER2 expression and ERBB2 amplification. Despite being operationally defined, HLBCs constitute a significant portion of breast cancers, more Figure A.8: The BioLunar report, produced by Biowork- flow for Scenario 1   Cancer type:                Breast Cancer   Sample size: 27   Cancer type: breast cancer   Biopsy site: primary site   Center:   Informed consent:   Sample sent: Export report Analysis run date: 2024-02-24 Pipeline version: v2.0. details   Context:   Breast cancer (BC) presents a significant global health challenge, with its incidence steadily rising and mortality rates remaining high. Its heterogeneous nature complicates treatment strategies, contributing to issues like recurrence and drug resistance. more Figure A.9: The BioLunar report, produced by Biowork- flow for Scenario 2.
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Nucleic Acids Research , 2025, 53 , D730–D737 https://doi.org/10.1093/nar/gkae1113 Advance access publication date: 18 November 2024 Database issue The STRING database in 2025: protein networks with directionality of regulation Damian Szklarczyk 1 , 2 , Katerina Nastou 3 , Mik aela K outrouli 3 , Rebecca Kirsch 3 , F ar rokh Mehryary 4 , Radja Hachilif 1 , 2 , Dewei Hu 3 , Matteo E. Peluso 1 , 2 , Qingyao Huang 1 , 2 , Tao Fang 1 , 2 , Nadezhda T. Doncheva 3 , Sampo Pyysalo 4 , P eer Bor k 5 , 6 , 7 , * , Lars J. Jensen 3 , * and Christian von Mering 1 , 2 , * 1 Department of Molecular Life Sciences, University of Zurich, Winterthurerstrasse 190, 8057 Zurich, Switzerland 2 SIB Swiss Institute of Bioinformatics, Amphipôle, Quartier UNIL-Sorge, 1015 Lausanne, Switzerland 3 Novo Nordisk Foundation Center for Protein Research, University of Copenhagen, Blegdamsvej 3B, 2200 Copenhagen N, Denmark 4 TurkuNLP Lab, Department of Computing, University of Turku, Vesilinnantie 5, 20014 Turku, Finland 5 Structural and Computational Biology Unit, European Molecular Biology Laboratory, Meyerhofstrasse 1, 69117 Heidelberg, Germany 6 Max Delbrück Centre for Molecular Medicine, Robert-Rössle-Strasse 10, 13125 Berlin, Germany 7 Department of Bioinformatics, Biozentrum, University of Würzburg, Am Hubland, 97074 Würzburg, Germany * To whom correspondence should be addressed. Tel: +49 6221 387 8526; Fax: +49 6221 387 517; Email: bork@embl.de Correspondence may also be addressed to Lars J. Jensen. Email: lars.juhl.jensen@cpr.ku.dk Correspondence may also be addressed to Christian von Mering. Tel: +41 44 6353147; Fax: +41 44 6356864; Email: mering@imls.uzh.ch Abstract Proteins cooperate, regulate and bind each other to achie v e their functions. Understanding the comple x netw ork of their interactions is essential f or a sy stems-le v el description of cellular processes. T he S TRING database compiles, scores and integrates protein–protein association informa- tion drawn from experimental assays, computational predictions and prior knowledge. Its goal is to create comprehensive and objective global networks that encompass both ph y sical and functional interactions. A dditionally, S TRING pro vides supplementary tools such as network cluster- ing and pathw a y enrichment analy sis. T he latest v ersion, S TRING 12.5, introduces a ne w ‘regulatory netw ork’, f or which it gathers evidence on the type and directionality of interactions using curated pathw a y databases and a fine-tuned language model parsing the literature. This update enables users to visualize and access three distinct network types—functional, ph y sical and regulatory —separately, each applicable to distinct research needs. In addition, the pathw a y enrichment detection functionality has been updated, with better false discovery rate corrections, redundancy filtering and impro v ed visual displa y s. T he resource now also offers improved annotations of clustered networks and provides users with do wnloadable netw ork embeddings, which f acilitate the use of S TRING netw orks in machine learning and allow cross-species transfer of protein information. The STRING database is available online at https:// string-db.org/ . Gr aphical abstr act Received: September 15, 2024. Revised: October 18, 2024. Editorial Decision: October 21, 2024. Accepted: October 29, 2024 © The Author(s) 2024. Published by Oxford University Press on behalf of Nucleic Acids Research. This is an Open Access article distributed under the terms of the Creative Commons Attribution License (https: // creativecommons.org / licenses / by / 4.0 / ), which permits unrestricted reuse, distribution, and reproduction in any medium, provided the original work is properly cited. Downloaded from https://academic.oup.com/nar/article/53/D1/D730/7903368 by guest on 11 January 2026
{ "author": "Szklarczyk Damian, Nastou Katerina, Koutrouli Mikaela, Kirsch Rebecca, Mehryary Farrokh, Hachilif Radja, Hu Dewei, Peluso Matteo E., Huang Qingyao, Fang Tao, Doncheva Nadezhda T., Pyysalo Sampo, Bork Peer, Jensen Lars J., vonMering Christian", "creationDate": "D:20241230125932+05'30'", "creationdate": "2024-12-30T12:59:32+05:30", "creator": "OUP", "file_path": "/home/donbr/open-biosciences/biosciences-research/data/raw/gkae1113.pdf", "format": "PDF 1.4", "keywords": "", "modDate": "D:20260111010257+00'00'", "moddate": "2026-01-11T01:02:57+00:00", "page": 0, "producer": "Acrobat Distiller 24.0 (Windows); modified using iTextSharp 5.5.10 ©2000-2016 iText Group NV (AGPL-version); modified using iTextSharp.LGPLv2.Core 3.7.4.0", "source": "/home/donbr/open-biosciences/biosciences-research/data/raw/gkae1113.pdf", "subject": "DOI: 10.1093/nar/gkae1113 , 53, Database issue, 18-11-2024. Abstract: Proteins cooperate, regulate and bind each other to achieve their functions. Understanding the complex network of their interactions is essential for a systems-level description of cellular processes. The STRING database compiles, scores and integrates protein&ndash;protein association information drawn from experimental assays, computational predictions and prior knowledge. Its goal is to create comprehensive and objective global networks that encompass both physical and functional interactions. Additionally, STRING provides supplementary tools such as network clustering and pathway enrichment analysis. The latest version, STRING 12.5, introduces a new &lsquo;regulatory network&rsquo;, for which it gathers evidence on the type and directionality of interactions using curated pathway databases and a fine-tuned language model parsing the literature. This update enables users to visualize and access three distinct network types&mdash;functional, physical and regulatory&mdash;separately, each applicable to distinct research needs. In addition, the pathway enrichment detection functionality has been updated, with better false discovery rate corrections, redundancy filtering&nbsp;and improved visual displays. The resource now also offers improved annotations of clustered networks and provides users with downloadable network embeddings, which facilitate the use of STRING networks in machine learning and allow cross-species transfer of protein information. The STRING database is available online at https:&sol;&sol;string-db.org&sol;.", "title": "The STRING database in 2025: protein networks with directionality of regulation", "total_pages": 8, "trapped": "" }
Nucleic Acids Research , 2025, Vol. 53, Database issue D 731 Introduction The function of every living cell is primarily governed by a complex network of interacting proteins, with each protein’s role determined not only by its molecular activities but also by its position within this network ( 1 ,2 ). Connected proteins work together to contribute to common biological processes through various interaction types, such as physical binding, genetic interactions and regulatory influences. These interac- tions can collectively be categorized as functional associa- tions , which serve as fundamental operational units within bi- ological systems. Unraveling protein networks in their various modalities remains a significant research focus. Consequently, numerous databases have been developed over the years to meet specific research needs. These range from carefully cu- rated pathway databases such as Reactome ( 3 ) and KEGG ( 4 ), to databases of experimental interaction evidence curated from literature such as BioGRID ( 5 ), IntAct ( 6 ) and MINT ( 7 ), and to those centered on specific modes of interactions, including Complex Portal for protein co-complexes ( 8 ) and SIGNOR for regulatory interactions ( 9 ). Finally, composite databases such as STRING ( 10 ), GeneMANIA ( 11 ), FunCoup ( 12 ) and HumanNet ( 13 ) not only incorporate data from these sources but also employ an array of computational methods to predict additional associations, striving to provide the most comprehensive views of the interactome. Among these databases, STRING is notable for its many sources of evidence, its robust scoring system, user-friendly interface and comprehensive suite of enrichment features. It is dedicated to assembling a broad set of associations among proteins for the complete proteomes of thousands of organ- isms across all domains of life. STRING charts interactions ranging from highly confident, well-documented associations to more speculative ones, which are crucial for exploratory and computational analyses. The scoring system ensures that data from diverse sources—including automatic text mining, high- and low-throughput experimental data, and compu- tational predictions—are directly comparable and weighted consistently, regardless of their origin. Additionally, STRING extends and maps networks across species by predicting in- terologs, thereby broadening the scope of functional associa- tions to encompass a diverse range of organisms. This includes uncultured novel bacterial species derived from metagenomic samples, and even unpublished proteomes uploaded by users. The development of interaction databases has so far been a trade-off between detailing the exact mode of an interaction— whether physical or functional, signaling or structural, stable or transient—and maintaining a comprehensive set of interac- tions. Given the limitations of the available data and method- ologies, STRING has historically focused on broadly defined functional associations, which provided the most useful set of interactions for proteome-wide analysis. While these net- works continue to perform well in downstream tasks ( 14 ,15 ), they often lack fine-grained resolution, leaving more special- ized databases better equipped to offer detailed insights into specific interactions if needed. However, the growing vol- ume of data and advancements in text-mining technologies have since enabled the composite, general-purpose databases to catch up, offering more detailed maps of the interac- tion space. Recently, the STRING database introduced a co- complex (physical) interaction network mode, which details multi-protein assemblies. With the latest update, the database now also includes the largest set of regulatory (directed) inter- actions, highlighting the flow of information within cells. This major enhancement greatly expands the analytical capabilities of the database, enabling new types of studies that were not previously possible with nondirected interaction networks. Interaction and pathway databases are frequently used to gain insights into the functional context of individual pro- teins or to understand the biological organization of an entire protein dataset. Many of these databases enhance the inter- pretation of protein datasets beyond simple interaction anno- tations by incorporating an enrichment analysis toolset. This type of analysis involves comparing observed data against ex- pected distributions, enabling researchers to identify statisti- cally significant features or patterns. Enrichment tools typi- cally utilize established ontologies and annotations, such as Gene Ontology ( 16 ), MSigDB hallmark sets ( 17 ) or OMIM ( 18 ). A few databases also leverage their annotated datasets to help understand the functional context of a user’s input. For example, KEGG ( 4 ) employs pathway and module ab- stractions to organize genes into datasets for overrepresenta- tion analysis, while Reactome maps interconnected proteins into hierarchically nested modules. Uniquely, STRING uses an unsupervised procedure to hierarchically precluster its en- tire proteome-wide networks into functional modules. Incor- porating these network-derived gene sets into the enrichment analysis facilitates the identification of novel modules, espe- cially in areas of the proteome where manual curation has been less comprehensive so far. Database content The basic interaction scope in STRING is that of a ‘functional association’ between pairs of proteins. A functional associa- tion is defined as a contribution of two non-identical proteins to a common function ( 19 ,20 ). This can take many forms; functionally associated proteins can be in physical proxim- ity to each other, regulate each other, exhibit genetic epistasis or even work antagonistically (as long as this occurs in the context of a common function). For the purpose of defining functional associations, the concept of a common function is crucial, but difficult to define—it should broadly be thought of as corresponding to the notion of a ‘pathway’ or ‘function module’. In practice, STRING roughly follows the functional granularity of the pathway maps in the KEGG database ( 4 ). From the set of all functional associations in STRING, sub- sets are derived that are more specifically annotated with re- gard to their mechanism of association. Currently, two such more specific subsets are implemented: ‘physical’ and ‘regula- tory’. The physical mode refers to pairs of proteins that either bind directly or are at least subunits of the same complex ( 21 ). The regulatory mode refers to associated protein pairs that are known to regulate each other’s activity in at least one direc- tion. This mode is described in more detail further below; for the first time in STRING, such connections are annotated not only with confidence scores but also with a directionality. All protein–protein associations in STRING are annotated with ‘confidence scores’. These scores are fully precomputed; they scale between 0 and 1 and describe the estimated like- lihood of a postulated association being correct, given the available evidence. Separate confidence scores are provided for physical and regulatory modes—these scores provide es- timates of the likelihood that the proposed association is tak- ing place and is indeed of the postulated type. The two spe- Downloaded from https://academic.oup.com/nar/article/53/D1/D730/7903368 by guest on 11 January 2026
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D 732 Nucleic Acids Research , 2025, Vol. 53, Database issue cific network modes are fully consistent with the functional network, such that if an interaction is present in either the physical or regulatory network, it will, by definition, also be present in the full functional association network (with an equal or higher confidence score). To compute the various con- fidence scores, the available evidence supporting a given inter- action is first separated into ‘evidence channels’, by the type of evidence. For the basic functional association confidence score, seven such channels are used. These include three that are based on genomic context predictions—neighborhood, fu- sion and gene co-occurrence—as well as channels dedicated to co-expression, experimental data, curated databases and text mining. The genomic context channels focus on associations in- ferred from genome sequences alone, detecting evolution- ary patterns such as gene proximity, fusion events and co- occurrence across different organisms. For instance, the neigh- borhood channel assigns an association score to gene pairs that are located close to each other on the chromosome in the same orientation (in prokaryotic genomes) ( 22 ). The fu- sion channel identifies open reading frames that result from gene fusion events ( 23 ), while the co-occurrence channel ex- amines whether genes have a nontrivial but shared distribu- tion across genomes ( 24 ), implying a shared history of hori- zontal transfers, losses or duplication events and thus likely related functions. The co-expression channel compiles data from gene expression studies, analyzing both transcript and protein abundances across various conditions. By comparing expression profiles, it identifies gene pairs with similar expres- sion patterns, suggesting functional linkage ( 10 ,25 ). The ex- periments channel aggregates interaction evidence from labo- ratory assays, including biochemical, biophysical and genetic assays. Data are imported from primary repositories such as BioGRID ( 5 ) and the IMEx consortium ( 26 ), and the confi- dence scores are estimated by globally benchmarking the accu- racy of annotated experimental techniques, as well as within- dataset performance and consistency for the case of high- throughput experiments ( 10 ). The database channel is based on well-described, curated protein–protein associations from expert-compiled resources, such as KEGG ( 4 ), Reactome ( 3 ) and Gene Ontology Complexes ( 16 ). These resources provide well-established pathways, complexes and functional relation- ships, offering a high level of reliability. Unlike other chan- nels, the database channel assigns a uniformly high confidence score to associations, reflecting their established nature. Fi- nally, the text-mining channel utilizes a large corpus of sci- entific literature, including PubMed abstracts and full-text ar- ticles, to identify co-mentions of protein names ( 27 ). By statis- tically analyzing the frequency of these co-mentions in various textual contexts, this channel uncovers potential associations that may not be evident from structured data alone. In addi- tion, for the more specific association modes, dedicated large language models are employed to detect sentences supporting either physical ( 21 ) or regulatory associations (see below). For each evidence channel, the available interaction evi- dence is translated into a confidence score by first quantifying the evidence using channel-specific metrics and then convert- ing these metrics into likelihoods using calibration curves de- rived from prior knowledge (from pathway-map memberships in KEGG). For the more specific association modes ‘physical’ and ‘regulatory’, only channels and evidence that are appli- cable to these modes are considered. After this, all channel scores that have been computed for a given protein pair in a given organism are transferred onto related protein pairs in other organisms, based on the ‘interolog’ concept ( 27 ,28 ). Lastly, a final, combined confidence score is computed by inte- grating the channel-specific subscores probabilistically, under the assumption that evidence in different channels is largely independent. Users of STRING can directly rely on this com- bined score for browsing and filtering networks, or they can alternatively customize their analyses by enabling or disabling specific channels separately, after which the combined score is recomputed according to their chosen settings. All primary evidence underlying a given interaction can be inspected interactively, in dedicated evidence viewers on- line. Furthermore, accessory information is available for each protein, such as its three-dimensional structure, do- main composition, annotation and cross-references. Apart from its core protein-network functionality, STRING also implements features that allow extensive analysis of up- loaded user data. These include functional enrichment de- tection ( 29 ), experimental bias detection ( 21 ), homology searches, and clustering and organizing large query pro- tein sets. All data contained in STRING are freely avail- able for download, under a Creative Commons BY 4.0 li- cense. Apart from its website, STRING can also be accessed via a dedicated Cytoscape plugin ( 30 ), through an applica- tion programming interface (API) ( 31 ), as well as through an R / Bioconductor package ( https://www.bioconductor.org/ packages/ release/ bioc/ html/ STRINGdb.html ). Regulat ory netw orks Molecular networks, like those available through STRING, have become invaluable tools in biomedical research, offering powerful insights into how molecules work together within cells. The functional association networks, while useful for many applications, do not specify the interaction type (e.g. complex formation or transcriptional regulation) nor the di- rection of interaction (i.e. who regulates whom). Constructing networks with more detailed interaction types and directions can significantly expand their utility, particularly for building mathematical models of biological systems or for making log- ical inferences. The directionality of interactions is essential for interpretation of omics data, which typically reveals more about downstream effects of conditions such as diseases than it does about the upstream events that led to it and thus could be targeted therapeutically. To incorporate interactions with specific directionality, sign and type into STRING, we leveraged the novel dataset and deep learning-based relation extraction method described in RegulaTome ( 32 ). The RegulaTome corpus provides a rich and diverse dataset that includes 16 961 relations between 54 951 entities annotated across over 2500 documents. This corpus is utilized to train a deep learning-based method used to annotate and extract the regulatory events throughout the entire available literature. On top of undirected physical interactions—which are al- ready included in STRING—the following types of directed interactions can be extracted using the system developed for RegulaTome: Regulation , with the signed subtypes of Posi- tive Regulation and Negative Regulation , Regulation of Gene Expression , Regulation of Degradation and Catalysis of Post- Translational Modifications , covering six subtypes: Catalysis of Small Protein Conjugation , Catalysis of Small Protein Re- moval , Catalysis of Phosphorylation , Catalysis of Dephospho- Downloaded from https://academic.oup.com/nar/article/53/D1/D730/7903368 by guest on 11 January 2026
{ "author": "Szklarczyk Damian, Nastou Katerina, Koutrouli Mikaela, Kirsch Rebecca, Mehryary Farrokh, Hachilif Radja, Hu Dewei, Peluso Matteo E., Huang Qingyao, Fang Tao, Doncheva Nadezhda T., Pyysalo Sampo, Bork Peer, Jensen Lars J., vonMering Christian", "creationDate": "D:20241230125932+05'30'", "creationdate": "2024-12-30T12:59:32+05:30", "creator": "OUP", "file_path": "/home/donbr/open-biosciences/biosciences-research/data/raw/gkae1113.pdf", "format": "PDF 1.4", "keywords": "", "modDate": "D:20260111010257+00'00'", "moddate": "2026-01-11T01:02:57+00:00", "page": 2, "producer": "Acrobat Distiller 24.0 (Windows); modified using iTextSharp 5.5.10 ©2000-2016 iText Group NV (AGPL-version); modified using iTextSharp.LGPLv2.Core 3.7.4.0", "source": "/home/donbr/open-biosciences/biosciences-research/data/raw/gkae1113.pdf", "subject": "DOI: 10.1093/nar/gkae1113 , 53, Database issue, 18-11-2024. Abstract: Proteins cooperate, regulate and bind each other to achieve their functions. Understanding the complex network of their interactions is essential for a systems-level description of cellular processes. The STRING database compiles, scores and integrates protein&ndash;protein association information drawn from experimental assays, computational predictions and prior knowledge. Its goal is to create comprehensive and objective global networks that encompass both physical and functional interactions. Additionally, STRING provides supplementary tools such as network clustering and pathway enrichment analysis. The latest version, STRING 12.5, introduces a new &lsquo;regulatory network&rsquo;, for which it gathers evidence on the type and directionality of interactions using curated pathway databases and a fine-tuned language model parsing the literature. This update enables users to visualize and access three distinct network types&mdash;functional, physical and regulatory&mdash;separately, each applicable to distinct research needs. In addition, the pathway enrichment detection functionality has been updated, with better false discovery rate corrections, redundancy filtering&nbsp;and improved visual displays. The resource now also offers improved annotations of clustered networks and provides users with downloadable network embeddings, which facilitate the use of STRING networks in machine learning and allow cross-species transfer of protein information. The STRING database is available online at https:&sol;&sol;string-db.org&sol;.", "title": "The STRING database in 2025: protein networks with directionality of regulation", "total_pages": 8, "trapped": "" }
Nucleic Acids Research , 2025, Vol. 53, Database issue D 733 Figure 1. Illustration of the new ‘regulatory network’ mode in STRING, where the network edges visually indicate the direction, confidence and sources of each regulatory interaction. Clicking on an edge within the network brings up a pop-up window with a detailed o v ervie w of the a v ailable e vidence and the annotated type of regulation. Users can further explore the data behind the interactions by accessing the specific evidence viewers linked within the pop-up, including the text-mining evidence viewer. This viewer presents excerpts from literature pertaining to the inspected interactions, as well as an automatically generated large language model summary of the presented e x cerpts. rylation , Catalysis of Small Molecule Conjugation and Cataly- sis of Small Molecule Removal . To enhance the interoperabil- ity of the annotations, these chosen relation types align with Gene Ontology ( 32 ). To identify and classify a wide array of interaction types, we fine-tuned the RoBERT a-large-PM-M3-V oc language model, a transformer-based model well suited for biological tasks. This model was trained on the RegulaTome dataset for multi- label extraction of the directed, typed and signed interactions mentioned above, and achieved an average F 1 score of 73.5% (with a precision of 75.2% and a recall of 71.8%) on a held- out test set, although the exact performance varied across dif- ferent types of relationships. This level of accuracy is substan- tial, considering the complexity of the underlying task. Using this model, we processed over 1.2 billion sentence-level pairs extracted from all available PubMed abstracts and PMC Open Access full-text documents, and assigned 3.5% of them ( ∼43 million, of which ∼18 million in human) with at least one positive label indicating directed (72.9%) or signed (33.1%) relationships among the proteins. Furthermore, we made use of the existing curated knowledge of regulatory interactions in the database channel, by parsing regulatory information from the SIGNOR, KEGG and Reactome databases. To integrate these interactions into STRING, we followed a similar score aggregation and benchmarking approach as for physical interactions ( 21 ). Here, we perform benchmark- ing in five categories for which we can derive gold stan- dard datasets of known human regulatory interactions from SIGNOR: regulation , upregulation , downregulation , tran- scriptional regulation and phosphorylation . The resulting calibration functions are then applied to extracted regula- tions of all types to produce the final regulatory confidence scores. The new regulatory network has been seamlessly integrated into the existing STRING framework, complementing the ‘functional’ and ‘physical’ network types. Directional edges within this network are visually represented by arrows, depict- ing both bidirectional and unidirectional relationships. The network visualization can show varying confidence levels be- tween directions (in the ‘confidence’ view) or the specific di- rectionality of the sources (in the ‘evidence’ view). The user interface retains its intuitive design, enabling users to access the type of regulatory event and the evidence for the interac- tion by clicking on the edge (Figure 1 ). Additionally, all API functions have been updated to fully support the new net- work type, which can be accessed by specifying the parameter netw or k_type = regulatory in the API call. Expanded co-expression networks The co-expression channel in STRING identifies genes with similar expression profiles across various tissues and condi- tions, revealing their involvement in shared biological pro- cesses. It compiles data from large-scale gene expression stud- ies, analyzing transcript and protein levels to detect gene pairs with coordinated expression. This method highlights poten- tial functional relationships between genes. Importantly, the analysis is free from study bias, as it directly uses raw omics data instead of relying on manually curated or experimental datasets, providing a more precise and objective view of gene interactions. For the upcoming version 12.5, we are expanding the co-expression channel by generating additional networks through the application of FAVA (Functional Association of Variational Autoencoders) ( 25 ) on a wider range of single- cell RNA sequencing data. Specifically, we are incorporating single-cell data from the cellxgene Atlas ( 33 ) to enhance the human and mouse networks and from the EBI Single Cell Ex- pression Atlas to extend coverage to more model organisms ( 34 ). This expansion will provide more detailed, organism- specific co-expression maps, enabling more precise identifi- cation of gene co-expression patterns and cross-species com- parisons, particularly for development, immune response and disease-related gene expression dynamics. Impro v ed enric hment analysis Gene set enrichment analysis is a critical component of high- throughput biological studies, providing key insights into the biological characteristics of datasets. This analysis is pivotal in identifying essential aspects such as biological processes, path- ways and phenotypes, which are crucial for understanding the biological implications of experiments. Typically, overrepre- sentation analysis is employed, wherein a subset of data ex- ceeding a specific cutoff (empirical or customary) is compared against an experimental background. By default, STRING Downloaded from https://academic.oup.com/nar/article/53/D1/D730/7903368 by guest on 11 January 2026
{ "author": "Szklarczyk Damian, Nastou Katerina, Koutrouli Mikaela, Kirsch Rebecca, Mehryary Farrokh, Hachilif Radja, Hu Dewei, Peluso Matteo E., Huang Qingyao, Fang Tao, Doncheva Nadezhda T., Pyysalo Sampo, Bork Peer, Jensen Lars J., vonMering Christian", "creationDate": "D:20241230125932+05'30'", "creationdate": "2024-12-30T12:59:32+05:30", "creator": "OUP", "file_path": "/home/donbr/open-biosciences/biosciences-research/data/raw/gkae1113.pdf", "format": "PDF 1.4", "keywords": "", "modDate": "D:20260111010257+00'00'", "moddate": "2026-01-11T01:02:57+00:00", "page": 3, "producer": "Acrobat Distiller 24.0 (Windows); modified using iTextSharp 5.5.10 ©2000-2016 iText Group NV (AGPL-version); modified using iTextSharp.LGPLv2.Core 3.7.4.0", "source": "/home/donbr/open-biosciences/biosciences-research/data/raw/gkae1113.pdf", "subject": "DOI: 10.1093/nar/gkae1113 , 53, Database issue, 18-11-2024. Abstract: Proteins cooperate, regulate and bind each other to achieve their functions. Understanding the complex network of their interactions is essential for a systems-level description of cellular processes. The STRING database compiles, scores and integrates protein&ndash;protein association information drawn from experimental assays, computational predictions and prior knowledge. Its goal is to create comprehensive and objective global networks that encompass both physical and functional interactions. Additionally, STRING provides supplementary tools such as network clustering and pathway enrichment analysis. The latest version, STRING 12.5, introduces a new &lsquo;regulatory network&rsquo;, for which it gathers evidence on the type and directionality of interactions using curated pathway databases and a fine-tuned language model parsing the literature. This update enables users to visualize and access three distinct network types&mdash;functional, physical and regulatory&mdash;separately, each applicable to distinct research needs. In addition, the pathway enrichment detection functionality has been updated, with better false discovery rate corrections, redundancy filtering&nbsp;and improved visual displays. The resource now also offers improved annotations of clustered networks and provides users with downloadable network embeddings, which facilitate the use of STRING networks in machine learning and allow cross-species transfer of protein information. The STRING database is available online at https:&sol;&sol;string-db.org&sol;.", "title": "The STRING database in 2025: protein networks with directionality of regulation", "total_pages": 8, "trapped": "" }
D 734 Nucleic Acids Research , 2025, Vol. 53, Database issue uses the whole genome / proteome background; however, it is recommended that users provide a custom background list representing a more realistic universe of genes / proteins detected by their assay ( 35 ). Alternatively, the entire sorted dataset can be analyzed using gene set enrichment analysis to determine whether the distribution of genes at the top, bottom or both ends deviates significantly from what would be ex- pected by chance. STRING supports both of these enrichment methods. The most recent update to STRING introduces vari- ous improvements, including a new false discovery rate (FDR) correction, enhanced filtering options and a new graphical vi- sualization. FDR correction In its analyses, STRING performs a test for each path- way (‘term’) within a given pathway collection (‘category’). Like many other enrichment tools, STRING employs the Benjamini–Hochberg correction ( 36 ) to adjust for multiple hypothesis testing. This statistical correction is essential for managing the FDR effectively. However, the larger the term count and the greater the diversity of the terms, the higher the required correction and the lower the chance of exceed- ing the alpha level (significance threshold) for a given term. One method used to address this issue involves restricting the analysis to a subset of terms, such as ‘GO slims’ that focus on broad, high-level terms with a simplified ontology struc- ture ( 16 ). However, this approach has some disadvantages: it might exclude terms particularly relevant to a tested gene set, it may not cover all functions by design and it is mainly ap- plicable to hierarchical classification systems such as the Gene Ontology. STRING utilizes several hierarchical ontologies, such as the Brenda Tissue Ontology, Disease Ontology, Human Phe- notype Ontology and the aforementioned Gene Ontology; they typically contain many more leaf-ward nodes than nodes closer to the root. The parental nodes must contain all the genes included in the child terms, creating an imbalance in the ontology with many more smaller terms and fewer larger terms. Removing smaller terms can substantially increase sta- tistical power . However , simply eliminating all small terms could negatively impact discoverability, especially for small query gene sets where the smaller terms might be the most rel- evant for biological interpretation. The ontologies are usually large, with > 10 000 terms (and therefore tests) in the Biolog- ical Process branch of the Gene Ontology tree. Such strong corrections have the effect that for larger query gene sets, or for backgrounds that cover only part of the proteome, it might not be statistically possible to detect enrichments for a subset of the terms; these can therefore be removed from considera- tion. In its new version, STRING takes a more flexible approach—by only testing terms that have a priori any statis- tical potential for enrichment. This decision is based on sev- eral parameters: the size of the term, the size of the query set, the background size and the number of tests conducted. By calculating the smallest and largest term sizes that could the- oretically be enriched given the FDR correction, STRING can determine which terms are viable for inclusion in the anal- ysis. Terms outside these bounds are excluded from testing. This methodical exclusion significantly enhances the statisti- cal power of the enrichment analysis, without omitting any terms that could be enriched. For smaller query sets, this strat- egy will maintain all terms, as even the smallest tested sets (term size = 2) might still be theoretically enriched. How- ever , for larger , less specific sets or for tests against a cus- tom smaller background, this approach markedly improves the statistical power, enabling STRING to perform more fo- cused analyses without the drawbacks of increased false posi- tive rates or omitting potentially biologically interesting terms. Although this method leverages the characteristics of ontolo- gies, it does not require a hierarchy (parent–child relationships between the terms) to function and can be applied to any pathway / term collection with an imbalance of small terms, such as PMID-derived gene sets and STRING neighborhood clusters. Term filtering One key insight from our online user surveys is that the of- ten overwhelming number of enrichment results can make it challenging to discern the most relevant enriched terms. We have thus enhanced the display of enrichment results, allow- ing users to filter and sort them based on (i) FDR, (ii) strength, (iii) signal, (iv) term size and (v) term similarity. The newest addition to our filtering options are the ‘term similarity’ and ‘signal’ filters. The ‘term similarity’ filter uses the Jaccard index to measure the similarity between the gene sets of terms within the same category. The method proceeds by sorting the terms according to their P -values, prioritizing those with the strongest statistical significance. The algorithm then iterates through the list of terms; any term that exhibits a similarity exceeding a predetermined, user-defined threshold relative to terms already included in the results is subsequently excluded. This approach requires no additional information beyond what is already available in the dataset and can be uniformly applied across all categories of terms. The ‘signal’ filter is defined as a weighted harmonic mean between the ra- tio of observed to expected gene occurrences in an enriched term and its −log(FDR), respectively. This filter balances the impact of both metrics and provides a more intuitive ordering of enriched terms and has been implemented as the default sorting criterion. These filters are designed to exclude potentially less relevant results, such as terms that are marginally significant, small or show significant overlap in terms of gene content. This en- hancement streamlines the search for relevant observations, helping users focus on the most meaningful results. Graphical visualization In response to user feedback requesting more intuitive, graph- ical visualizations of the detected enrichments, STRING now has adopted an interactive dot plot as a primary display method for enrichment analysis outcomes. We chose the dot plot for its clarity in representing enriched functions across three dimensions: (i) enrichment signal along the X -axis, (ii) the FDR indicated through the color coding of the dots and (iii) the term’s protein count in the network represented by the size of each dot. The terms (listed on the Y -axis) are ranked by their enrichment signal or, if grouped by similarity, by the enrichment signal of their group first (Figure 2 ). This visual- ization reflects all filter settings chosen by the user to manage the volume of results displayed. These plots can be addition- ally customized in the interface and the resulting graphs can be downloaded as PNG for immediate use or as SVG for further modifications. Downloaded from https://academic.oup.com/nar/article/53/D1/D730/7903368 by guest on 11 January 2026
{ "author": "Szklarczyk Damian, Nastou Katerina, Koutrouli Mikaela, Kirsch Rebecca, Mehryary Farrokh, Hachilif Radja, Hu Dewei, Peluso Matteo E., Huang Qingyao, Fang Tao, Doncheva Nadezhda T., Pyysalo Sampo, Bork Peer, Jensen Lars J., vonMering Christian", "creationDate": "D:20241230125932+05'30'", "creationdate": "2024-12-30T12:59:32+05:30", "creator": "OUP", "file_path": "/home/donbr/open-biosciences/biosciences-research/data/raw/gkae1113.pdf", "format": "PDF 1.4", "keywords": "", "modDate": "D:20260111010257+00'00'", "moddate": "2026-01-11T01:02:57+00:00", "page": 4, "producer": "Acrobat Distiller 24.0 (Windows); modified using iTextSharp 5.5.10 ©2000-2016 iText Group NV (AGPL-version); modified using iTextSharp.LGPLv2.Core 3.7.4.0", "source": "/home/donbr/open-biosciences/biosciences-research/data/raw/gkae1113.pdf", "subject": "DOI: 10.1093/nar/gkae1113 , 53, Database issue, 18-11-2024. Abstract: Proteins cooperate, regulate and bind each other to achieve their functions. Understanding the complex network of their interactions is essential for a systems-level description of cellular processes. The STRING database compiles, scores and integrates protein&ndash;protein association information drawn from experimental assays, computational predictions and prior knowledge. Its goal is to create comprehensive and objective global networks that encompass both physical and functional interactions. Additionally, STRING provides supplementary tools such as network clustering and pathway enrichment analysis. The latest version, STRING 12.5, introduces a new &lsquo;regulatory network&rsquo;, for which it gathers evidence on the type and directionality of interactions using curated pathway databases and a fine-tuned language model parsing the literature. This update enables users to visualize and access three distinct network types&mdash;functional, physical and regulatory&mdash;separately, each applicable to distinct research needs. In addition, the pathway enrichment detection functionality has been updated, with better false discovery rate corrections, redundancy filtering&nbsp;and improved visual displays. The resource now also offers improved annotations of clustered networks and provides users with downloadable network embeddings, which facilitate the use of STRING networks in machine learning and allow cross-species transfer of protein information. The STRING database is available online at https:&sol;&sol;string-db.org&sol;.", "title": "The STRING database in 2025: protein networks with directionality of regulation", "total_pages": 8, "trapped": "" }
Nucleic Acids Research , 2025, Vol. 53, Database issue D 735 Figure 2. Enrichment analysis interface from the STRING database website. Lef t: Sc hematic of the enrichment analysis tab highlighting various sections of the webpage. Top right: A zoomed-in view of the analysis table with two user-highlighted terms (colored lines). Bottom right: A visualization of the enrichment depicting the two terms highlighted in corresponding colors. Furthermore, a distinctive feature of STRING’s visualiza- tion is the incorporation of similarity groups, which visually group related terms on the plot, adding an additional dimen- sion to the enrichment results. These groups utilize computed similarities to cluster terms together, highlighting relationships between the terms and aiding the analysis. The clustering em- ploys the average linkage method based on the Jaccard index, with a user-specified cutoff applied to form groups that, on average, share the specified similarity. The groups are sorted by the maximum signal of their terms, with each term within a group subsequently sorted by its individual signal. This group- ing is visually highlighted on the right side of the plot. S TRING clust ering and g ene set descriptions STRING offers network clustering options for user-submitted gene lists. This feature visually connects nodes that are more interconnected to each other than to other nodes, reflecting the functional modularity of the user’s gene list. By grouping proteins that closely interact, clustering aids in the discovery of functional modules and facilitates hypothesis generation. Clustering is based on the connectivity between nodes, incor- porating edge weights (combined scores) reflecting the confi- dence level of the interactions. Only visible edges—those cor- responding to active channels with confidence higher than the user-specified cutoff—are considered, ensuring that clustering is consistent with the network being shown. Users have the choice of two different clustering algorithms. ‘ K -means clustering’ is implemented via the k -medoids algo- rithm [pyclustering package ( 37 )], allowing users to specify the desired number of clusters ( k ). This method forms clusters based on a distance matrix derived from the most probable paths between nodes. In case of multiple disconnected compo- nents within the network, STRING aims for a balanced distri- bution of cluster sizes by iteratively dividing the largest com- ponents first. In contrast, ‘MCL clustering’, implemented with standalone binaries (release 14-137), automatically generates natural divisions based on the network’s inherent structure. It utilizes the Markov cluster algorithm ( 38 ) to simulate stochas- tic flow in graphs, identifying groups of highly interconnected nodes as clusters based on their combined STRING scores. The user-controlled inflation parameter influences the granu- larity of the clustering. This method is particularly effective in capturing the true modular nature of biological networks, allowing for intuitive groupings that correspond to biological functions and relationships. Researchers often analyze the biological context of the ob- tained clusters to interpret and draw conclusions from net- work data. To aid this process, we have developed a novel gene set naming feature that significantly enhances the inter- pretability of cluster analyses. This feature automatically as- signs the best description for each cluster based on STRING’s robust statistical enrichment analysis, simplifying the often cumbersome task of manually interpreting cluster data. It ranks the enriched terms by their enrichment signal (see the ‘Improved enrichment analysis’ section), choosing the pri- mary, secondary and tertiary names among various enrich- ment categories. This prioritization ensures that the names re- flect the most statistically significant and biologically pertinent attributes of each cluster, providing a clear, immediate under- standing of its functional characteristics. The enrichment pri- marily draws from categories such as Gene Ontology Biolog- ical Processes, while incorporating a range of other categories for broader annotations. In case the enrichment analysis yields no significant results or if the cluster contains only one gene, STRING assigns canonical gene names as descriptions to en- Downloaded from https://academic.oup.com/nar/article/53/D1/D730/7903368 by guest on 11 January 2026
{ "author": "Szklarczyk Damian, Nastou Katerina, Koutrouli Mikaela, Kirsch Rebecca, Mehryary Farrokh, Hachilif Radja, Hu Dewei, Peluso Matteo E., Huang Qingyao, Fang Tao, Doncheva Nadezhda T., Pyysalo Sampo, Bork Peer, Jensen Lars J., vonMering Christian", "creationDate": "D:20241230125932+05'30'", "creationdate": "2024-12-30T12:59:32+05:30", "creator": "OUP", "file_path": "/home/donbr/open-biosciences/biosciences-research/data/raw/gkae1113.pdf", "format": "PDF 1.4", "keywords": "", "modDate": "D:20260111010257+00'00'", "moddate": "2026-01-11T01:02:57+00:00", "page": 5, "producer": "Acrobat Distiller 24.0 (Windows); modified using iTextSharp 5.5.10 ©2000-2016 iText Group NV (AGPL-version); modified using iTextSharp.LGPLv2.Core 3.7.4.0", "source": "/home/donbr/open-biosciences/biosciences-research/data/raw/gkae1113.pdf", "subject": "DOI: 10.1093/nar/gkae1113 , 53, Database issue, 18-11-2024. Abstract: Proteins cooperate, regulate and bind each other to achieve their functions. Understanding the complex network of their interactions is essential for a systems-level description of cellular processes. The STRING database compiles, scores and integrates protein&ndash;protein association information drawn from experimental assays, computational predictions and prior knowledge. Its goal is to create comprehensive and objective global networks that encompass both physical and functional interactions. Additionally, STRING provides supplementary tools such as network clustering and pathway enrichment analysis. The latest version, STRING 12.5, introduces a new &lsquo;regulatory network&rsquo;, for which it gathers evidence on the type and directionality of interactions using curated pathway databases and a fine-tuned language model parsing the literature. This update enables users to visualize and access three distinct network types&mdash;functional, physical and regulatory&mdash;separately, each applicable to distinct research needs. In addition, the pathway enrichment detection functionality has been updated, with better false discovery rate corrections, redundancy filtering&nbsp;and improved visual displays. The resource now also offers improved annotations of clustered networks and provides users with downloadable network embeddings, which facilitate the use of STRING networks in machine learning and allow cross-species transfer of protein information. The STRING database is available online at https:&sol;&sol;string-db.org&sol;.", "title": "The STRING database in 2025: protein networks with directionality of regulation", "total_pages": 8, "trapped": "" }
D 736 Nucleic Acids Research , 2025, Vol. 53, Database issue sure that clusters are easily identifiable. This functionality ex- tends beyond cluster analysis and is applicable to any gene set. As such, it is also available through an API, complementing our suite of other API methods. The new API function, named geneset_description , requires only a set of genes as input and automatically performs enrichment analysis. It returns up to three descriptions—primary, secondary and tertiary—based on relevance and availability, identical to the cluster naming in the user interface. These descriptions are filled sequentially, with the ‘primary’ always representing the most relevant term. One application of this API is in the stringApp Cytoscape plu- gin, which utilizes it to automatically overlay the assigned de- scriptions onto each network cluster after clustering is exe- cuted. Network and protein embeddings To facilitate the use of STRING in machine learning applica- tions, users can now directly download precomputed ProtT5 ( 39 ) sequence and cross-species protein network embeddings derived from STRING, for all eukaryotes, and utilize the em- beddings on their own labeled datasets. The embeddings en- code information from two aspects. Sequence embeddings can capture protein domains and shorter sequence motifs, while network embeddings can complement these with information on cellular organization such as protein complexes and path- ways that may not be evident from sequence data alone. While protein sequence embeddings are inherently compa- rable across species due to the universal nature of amino acid sequences, the primary obstacle to using network embeddings is ensuring that network embeddings from different species are directly comparable. This challenge arises because pro- tein networks are independent for each species, and tradi- tional network embedding methods are not designed to ad- dress cross-species comparability. The lack of comparability between network embeddings prevents the effective transfer of knowledge and findings across different species. To address this, we developed a method to align eukaryotic network em- beddings across species using orthologous relationships based on FedCoder ( 40 ). This alignment technique creates a uni- fied embedding space where proteins from different eukary- otic species can be directly compared. As a result, it enhances cross-species protein predictions, particularly in tasks such as subcellular localization and function prediction. In the protein machine learning field, having precomputed sequence embeddings and cross-species compatible network embeddings from STRING enables researchers to use these re- sources directly, eliminating the need to calculate embeddings themselves. This strategy not only reduces the energy footprint resulting from redundant calculations across research groups, but also enhances reproducibility in computational biology re- search promoting more sustainable and consistent scientific practices. Data availability The STRING database is freely available online at https:// string-db.org/. A c kno wledg ements The authors wish to thank Yan P. Yuan (EMBL Heidelberg) and Dandan Xue (University of Copenhagen) for IT support. Thomas Rattei (University of Vienna) is thanked for extensive computational work toward resolving orthology relations. We thank the CSC—IT Center for Science, Finland, for generous computational resources. Funding Swiss Institute of Bioinformatics; Novo Nordisk Founda- tion [NNF14CC0001, NNF20SA0035590]; European Molec- ular Biology Laboratory (EMBL Heidelberg); HORIZON EUROPE Marie Skłodowska-Curie Actions [101023676 to K.N.]; Academy of Finland [332844 to F.M. and S.P.]. Fund- ing for open access charge: University of Zurich. Conflict of interest statement None declared. References 1. Marsh, J.A. and Teichmann, S.A. (2015) Structure, dynamics, assembly, and evolution of protein complexes. Annu. Rev. Biochem., 84 , 551–575. 2. Cesnik, A. , Schaffer, L.V. , Gaur, I. , Jain, M. , Ideker, T. and Lundberg,E. (2024) Mapping the multiscale proteomic organization of cellular and disease phenotypes. Annu. Rev. Biomed. Data Sci., 7 , 369–389. 3. Milacic, M. , Beavers, D. , Conley, P. , Gong, C. , Gillespie, M. , Griss, J. , Haw, R. , Jassal, B. , Matthews, L. , May, B. , et al. (2024) The Reactome Pathway Knowledgebase 2024. Nucleic Acids Res., 52 , D672–D678. 4. Kanehisa, M. , Furumichi, M. , Sato, Y. , Kawashima, M. and Ishiguro-Watanabe,M. (2023) KEGG for taxonomy-based analysis of pathways and genomes. Nucleic Acids Res. , 51 , D587–D592. 5. Oughtred, R. , Stark, C. , Breitkreutz, B.-J. , Rust, J. , Boucher, L. , Chang, C. , Kolas, N. , O’Donnell, L. , Leung, G. , McAdam, R. , et al. (2019) The BioGRID interaction database: 2019 update. Nucleic Acids Res., 47 , D529–D541. 6. Del Toro, N. , Shrivastava, A. , Ragueneau, E. , Meldal, B. , Combe, C. , Barrera, E. , Perfetto, L. , How, K. , Ratan, P. , Shirodkar, G. , et al. (2022) The IntAct database: efficient access to fine-grained molecular interaction data. Nucleic Acids Res. , 50 , D648–D653. 7. Licata, L. , Briganti, L. , Peluso, D. , Perfetto, L. , Iannuccelli, M. , Galeota, E. , Sacco, F. , Palma, A. , Nardozza, A.P. , Santonico, E. , et al. (2012) MINT, the molecular interaction database: 2012 update. Nucleic Acids Res., 40 , D857–D861. 8. Meldal, B.H.M. , Perfetto, L. , Combe, C. , Lubiana, T. , Ferreira Cavalcante, J.V. , Bye-A-Jee, H. , Waagmeester, A. , Del-Toro, N. , Shrivastava, A. , Barrera, E. , et al. (2022) Complex Portal 2022: new curation frontiers. Nucleic Acids Res. , 50 , D578–D586. 9. Lo Surdo, P. , Iannuccelli, M. , Contino, S. , Castagnoli, L. , Licata, L. , Cesareni, G. and Perfetto, L. (2023) SIGNOR 3.0, the SIGnaling network open resource 3.0: 2022 update. Nucleic Acids Res., 51 , D631–D637. 10. Szklarczyk, D. , Kirsch, R. , Koutrouli, M. , Nastou, K. , Mehryary, F. , Hachilif, R. , Gable, A.L. , Fang, T. , Doncheva, N.T. , Pyysalo, S. , et al. (2023) The STRING database in 2023: protein–protein association networks and functional enrichment analyses for any sequenced genome of interest. Nucleic Acids Res. , 51 , D638–D646. 11. Warde-Farley, D. , Donaldson, S.L. , Comes, O. , Zuberi, K. , Badrawi, R. , Chao, P. , Franz, M. , Grouios, C. , Kazi, F. , Lopes, C.T. , et al. (2010) The GeneMANIA prediction server: biological network integration for gene prioritization and predicting gene function. Nucleic Acids Res. , 38 , W214–W220. 12. Persson, E. , Castresana-Aguirre, M. , Buzzao, D. , Guala, D. and Sonnhammer,E.L.L. (2021) FunCoup 5: functional association Downloaded from https://academic.oup.com/nar/article/53/D1/D730/7903368 by guest on 11 January 2026
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Nucleic Acids Research , 2025, Vol. 53, Database issue D 737 networks in all domains of life, supporting directed links and tissue-specificity. J. Mol. Biol., 433 , 166835. 13. Kim, C.Y. , Baek, S. , Cha, J. , Yang, S. , Kim, E. , Marcotte, E.M. , Hart, T. and Lee,I. (2022) HumanNet v3: an improved database of human gene networks for disease research. Nucleic Acids Res., 50 , D632–D639. 14. Huang, J.K. , Carlin, D.E. , Yu, M.K. , Zhang, W. , Kreisberg, J.F. , Tamayo, P. and Ideker, T. (2018) Systematic evaluation of molecular networks for discovery of disease genes. Cell Syst. , 6 , 484–495.e5. 15. Wright, S.N. , Colton, S. , Schaffer, L.V. , Pillich, R.T. , Churas, C. , Pratt, D. and Ideker, T. (2024) State of the interactomes: an evaluation of molecular networks for generating biological insights. bioRxiv doi: https:// doi.org/ 10.1101/ 2024.04.26.587073 , 29 April 2024, preprint: not peer reviewed. 16. Ontology Consortium, G. , Aleksander, S.A. , Balhoff, J. , Carbon, S. , Cherry, J.M. , Drabkin, H.J. , Ebert, D. , Feuermann, M. , Gaudet, P. , Harris, N.L. , et al. (2023) The Gene Ontology knowledgebase in 2023. Genetics , 224 , iyad031. 17. Liberzon, A. , Birger, C. , Thorvaldsdóttir, H. , Ghandi, M. , Mesirov, J.P. and Tamayo, P. (2015) The Molecular Signatures Database hallmark gene set collection. Cell Syst. , 1 , 417–425. 18. Amberger, J.S. , Bocchini, C.A. , Schiettecatte, F. , Scott, A.F. and Hamosh,A. (2015) OMIM.org: Online Mendelian Inheritance in Man (OMIM ®), an online catalog of human genes and genetic disorders. Nucleic Acids Res. , 43 , D789–D798. 19. Snel, B. , Bork, P. and Huynen, M.A. (2002) The identification of functional modules from the genomic association of genes. Proc. Natl Acad. Sci. U.S.A., 99 , 5890–5895. 20. Guala, D. , Ogris, C. , Müller, N. and Sonnhammer, E.L.L. (2020) Genome-wide functional association networks: background, data & state-of-the-art resources. Brief. Bioinform., 21 , 1224. 21. Szklarczyk, D. , Gable, A.L. , Nastou, K.C. , Lyon, D. , Kirsch, R. , Pyysalo, S. , Doncheva, N.T. , Legeay, M. , Fang, T. , Bork, P. , et al. (2021) The STRING database in 2021: customizable protein–protein networks, and functional characterization of user-uploaded gene / measurement sets. Nucleic Acids Res., 49 , D605–D612. 22. Mering,C.V. (2003) STRING: a database of predicted functional associations between proteins. Nucleic Acids Res. , 31 , 258–261. 23. Enright, A.J. , Iliopoulos, I. , Kyrpides, N.C. and Ouzounis, C.A. (1999) Protein interaction maps for complete genomes based on gene fusion events. Nature , 402 , 86–90. 24. Franceschini, A. , Lin, J. , von Mering, C. and Jensen, L.J. (2016) SVD-phy: improved prediction of protein functional associations through singular value decomposition of phylogenetic profiles. Bioinformatics , 32 , 1085–1087. 25. Koutrouli, M. , Nastou, K. , Piera Líndez, P. , Bouwmeester, R. , Rasmussen, S. , Martens, L. and Jensen, L.J. (2024) FAVA: high-quality functional association networks inferred from scRNA-seq and proteomics data. Bioinformatics , 40 , btae010. 26. Porras, P. , Barrera, E. , Bridge, A. , Del-Toro, N. , Cesareni, G. , Duesbury, M. , Hermjakob, H. , Iannuccelli, M. , Jurisica, I. , Kotlyar, M. , et al. (2020) Towards a unified open access dataset of molecular interactions. Nat. Commun., 11 , 6144. 27. Franceschini, A. , Szklarczyk, D. , Frankild, S. , Kuhn, M. , Simonovic, M. , Roth, A. , Lin, J. , Minguez, P. , Bork, P. , von Mering, C. , et al. (2013) STRING v9.1: protein–protein interaction networks, with increased coverage and integration. Nucleic Acids Res., 41 , D808–D815. 28. Yu, H. , Luscombe, N.M. , Lu, H.X. , Zhu, X. , Xia, Y. , Han, J .-D.J ., Bertin, N. , Chung, S. , V idal, M. and Gerstein, M. (2004) Annotation transfer between genomes: protein–protein interologs and protein–DNA regulogs. Genome Res. , 14 , 1107–1118. 29. Szklarczyk, D. , Gable, A.L. , Lyon, D. , Junge, A. , Wyder, S. , Huerta-Cepas, J. , Simonovic, M. , Doncheva, N.T. , Morris, J.H. , Bork, P. , et al. (2019) STRING v11: protein–protein association networks with increased coverage, supporting functional discovery in genome-wide experimental datasets. Nucleic Acids Res., 47 , D607–D613. 30. Doncheva, N.T. , Morris, J.H. , Gorodkin, J. and Jensen, L.J. (2019) Cytoscape StringApp: network analysis and visualization of proteomics data. J. Proteome Res., 18 , 623–632. 31. Jensen, L.J. , Kuhn, M. , Stark, M. , Chaffron, S. , Creevey, C. , Muller, J. , Doerks, T. , Julien, P. , Roth, A. , Simonovic, M. , et al. (2009) STRING 8—a global view on proteins and their functional interactions in 630 organisms. Nucleic Acids Res. , 37 , D412–D416. 32. Nastou, K. , Mehryary, F. , Ohta, T. , Luoma, J. , Pyysalo, S. and Jensen,L.J. (2024) RegulaTome: a corpus of typed, directed, and signed relations between biomedical entities in the scientific literature. Database , 2024 , baae095. 33. Megill, C. , Martin, B. , Weaver, C. , Bell, S. , Prins, L. , Badajoz, S. , McCandless, B. , Pisco, A.O. , Kinsella, M. , Griffin, F. , et al. (2021) cellxgene: a performant, scalable exploration platform for high dimensional sparse matrices. bioRxiv doi: https:// doi.org/ 10.1101/ 2021.04.05.438318 , 6 April 2021, preprint: not peer reviewed. 34. Moreno, P. , Fexova, S. , George, N. , Manning, J.R. , Miao, Z. , Mohammed, S. , Muñoz-Pomer, A. , Fullgrabe, A. , Bi, Y. , Bush, N. , et al. (2022) Expression Atlas update: gene and protein expression in multiple species. Nucleic Acids Res. , 50 , D129–D140. 35. T immons, J.A. , Szkop, K.J. and Gallagher, I.J. (2015) Multiple sources of bias confound functional enrichment analysis of global -omics data. Genome Biol. , 16 , 186. 36. Benjamini, Y. and Hochberg, Y. (1995) Controlling the false discovery rate: a practical and powerful approach to multiple testing. J. R. Stat. Soc. Ser. B Stat. Methodol., 57 , 289–300. 37. Novikov,A. (2019) PyClustering: data mining library. J. Open Source Softw., 4 , 1230. 38. Van Dongen,S. (2008) Graph clustering via a discrete uncoupling process. SIAM J. Matrix Anal. Appl., 30 , 121–141. 39. Elnaggar, A. , Heinzinger, M. , Dallago, C. , Rehawi, G. , Wang, Y. , Jones, L. , Gibbs, T. , Feher, T. , Angerer, C. , Steinegger, M. , et al. (2022) ProtTrans: toward understanding the language of life through self-supervised learning. IEEE Trans. Pattern Anal. Mach. Intell., 44 , 7112–7127. 40. Baumgartner, M. , Dell’Aglio, D. , Paulheim, H. and Bernstein, A. (2023) Towards the Web of Embeddings: integrating multiple knowledge graph embedding spaces with FedCoder. J. Web Semant., 75 , 100741. Received: September 15, 2024. Revised: October 18, 2024. Editorial Decision: October 21, 2024. Accepted: October 29, 2024 © The Author(s) 2024. Published by Oxford University Press on behalf of Nucleic Acids Research. This is an Open Access article distributed under the terms of the Creative Commons Attribution License (https: // creativecommons.org / licenses / by / 4.0 / ), which permits unrestricted reuse, distribution, and reproduction in any medium, provided the original work is properly cited. Downloaded from https://academic.oup.com/nar/article/53/D1/D730/7903368 by guest on 11 January 2026
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Research and Applications Improving large language model applications in biomedicine with retrieval-augmented generation: a systematic review, meta-analysis, and clinical development guidelines Siru Liu, PhD,1,2, Allison B. McCoy , PhD1, Adam Wright , PhD1,3 1Department of Biomedical Informatics, Vanderbilt University Medical Center, Nashville, TN 37212, United States, 2Department of Computer Science, Vanderbilt University, Nashville, TN 37212, United States, 3Department of Medicine, Vanderbilt University Medical Center, Nashville, TN 37212, United States Corresponding author: Siru Liu, PhD, Department of Biomedical Informatics, Vanderbilt University Medical Center, 2525 West End Ave #1475, Nashville, TN 37212, United States (siru.liu@vumc.org) Abstract Objective: The objectives of this study are to synthesize findings from recent research of retrieval-augmented generation (RAG) and large lan­ guage models (LLMs) in biomedicine and provide clinical development guidelines to improve effectiveness. Materials and Methods: We conducted a systematic literature review and a meta-analysis. The report was created in adherence to the Pre­ ferred Reporting Items for Systematic Reviews and Meta-Analyses 2020 analysis. Searches were performed in 3 databases (PubMed, Embase, PsycINFO) using terms related to “retrieval augmented generation” and “large language model,” for articles published in 2023 and 2024. We selected studies that compared baseline LLM performance with RAG performance. We developed a random-effect meta-analysis model, using odds ratio as the effect size. Results: Among 335 studies, 20 were included in this literature review. The pooled effect size was 1.35, with a 95% confidence interval of 1.19-1.53, indicating a statistically significant effect (P ¼ .001). We reported clinical tasks, baseline LLMs, retrieval sources and strategies, as well as evaluation methods. Discussion: Building on our literature review, we developed Guidelines for Unified Implementation and Development of Enhanced LLM Appli­ cations with RAG in Clinical Settings to inform clinical applications using RAG. Conclusion: Overall, RAG implementation showed a 1.35 odds ratio increase in performance compared to baseline LLMs. Future research should focus on (1) system-level enhancement: the combination of RAG and agent, (2) knowledge-level enhancement: deep integration of knowledge into LLM, and (3) integration-level enhancement: integrating RAG systems within electronic health records. Key words: large language model; retrieval augmented generation; systematic review; meta-analysis. Introduction Large language models (LLMs) have reported remarkable performance in question-answering, summarization, and text generation.1 Given this, researchers have explored its poten­ tial in biomedical areas.2 For example, several studies reported the ability of using LLM to answer patient mes­ sages,3 to analyze alert logic in clinical decision support,4 and to make discharge summaries more readable to patients.5 However, several challenges remain. LLMs are trained on fixed datasets, which restrict their knowledge to information available up to the training cut-off date. For example, GPT-4o’s training data only includes information up to October 2023, making it unable to respond accurately to findings that emerged afterward. LLM training datasets are also generally broad and lack the specif­ icity required for biomedical applications. Finally, not all sources used to train the LLMs are reliable and trustworthy. To address these limitations, researchers have performed fine-tuning and retrieval-augmented generation (RAG) tech­ niques. Fine-tuning can adapt LLMs to specific domains, but it is resource-intensive and does not allow for real-time updates. In contrast, RAG maintains the original LLM archi­ tecture while incorporating relevant context directly into queries, offering more flexibility and control. In addition, RAG’s unique advantage in biomedical applications lies in its ability to adapt to dynamic environments by delivering up-to- date information and efficiently integrating external knowl­ edge sources with high interpretability.6 Another limitation of using LLMs directly is the risk of hal­ lucination, where the model generates incorrect or fabricated information.7 To mitigate such issues, researchers have pro­ posed RAG as a solution that integrates up-to-date, relevant information, enhancing both the accuracy and reliability of LLM generated responses.8,9 For example, when ChatGPT Received: November 19, 2024; Revised: December 17, 2024; Editorial Decision: January 2, 2025; Accepted: January 3, 2025 © The Author(s) 2025. Published by Oxford University Press on behalf of the American Medical Informatics Association. This is an Open Access article distributed under the terms of the Creative Commons Attribution-NonCommercial License (https://creativecommons.org/licenses/ by-nc/4.0/), which permits non-commercial re-use, distribution, and reproduction in any medium, provided the original work is properly cited. For commercial re-use, please contact reprints@oup.com for reprints and translation rights for reprints. All other permissions can be obtained through our RightsLink service via the Permissions link on the article page on our site—for further information please contact journals.permissions@oup.com. Journal of the American Medical Informatics Association, 2025, 32(4), 605–615 https://doi.org/10.1093/jamia/ocaf008 Advance access publication 15 January 2025 Research and Applications Downloaded from https://academic.oup.com/jamia/article/32/4/605/7954485 by guest on 11 January 2026
{ "author": "", "creationDate": "D:20250322110116+05'30'", "creationdate": "2025-03-22T11:01:16+05:30", "creator": "Servigistics Arbortext Advanced Print Publisher 11.1.4667/W", "file_path": "/home/donbr/open-biosciences/biosciences-research/data/raw/ocaf008.pdf", "format": "PDF 1.5", "keywords": "large language model; retrieval augmented generation; systematic review; meta-analysis", "modDate": "D:20260111011327+00'00'", "moddate": "2026-01-11T01:13:27+00:00", "page": 0, "producer": "PDFlib+PDI 9.0.7p3 (C++/Win32); modified using iTextSharp.LGPLv2.Core 3.7.4.0", "source": "/home/donbr/open-biosciences/biosciences-research/data/raw/ocaf008.pdf", "subject": "Doi: 10.1093/jamia/ocaf008 Journal of the American Medical Informatics Association, 32, 4, 2025 Publication Date: 15/01/2025 Abstract Objective The objectives of this study are to synthesize findings from recent research of retrieval-augmented generation (RAG) and large language models (LLMs) in biomedicine and provide clinical development guidelines to improve effectiveness.Materials and Methods We conducted a systematic literature review and a meta-analysis. The report was created in adherence to the Preferred Reporting Items for Systematic Reviews and Meta-Analyses 2020 analysis. Searches were performed in 3 databases (PubMed, Embase, PsycINFO) using terms related to “retrieval augmented generation” and “large language model,” for articles published in 2023 and 2024. We selected studies that compared baseline LLM performance with RAG performance. We developed a random-effect meta-analysis model, using odds ratio as the effect size.Results Among 335 studies, 20 were included in this literature review. The pooled effect size was 1.35, with a 95% confidence interval of 1.19-1.53, indicating a statistically significant effect (P = .001). We reported clinical tasks, baseline LLMs, retrieval sources and strategies, as well as evaluation methods.Discussion Building on our literature review, we developed Guidelines for Unified Implementation and Development of Enhanced LLM Applications with RAG in Clinical Settings to inform clinical applications using RAG.Conclusion Overall, RAG implementation showed a 1.35 odds ratio increase in performance compared to baseline LLMs. Future research should focus on (1) system-level enhancement: the combination of RAG and agent, (2) knowledge-level enhancement: deep integration of knowledge into LLM, and (3) integration-level enhancement: integrating RAG systems within electronic health records.", "title": "Improving large language model applications in biomedicine with retrieval-augmented generation: a systematic review, meta-analysis, and clinical development guidelines", "total_pages": 11, "trapped": "" }
was asked about medications for peripheral artery disease patients without increased bleeding risk, it initially omitted low-dose rivaroxaban. After integrating retrieved text from the 2024 American College of Cardiology / American Heart Association Guideline for the Management of Lower Extrem­ ity Peripheral Artery Disease,10 the model correctly recom­ mended rivaroxaban. Several guidelines exist for evaluating Artificial Intelligence (AI) applications and LLMs in healthcare, including DECIDE-AI (Developmental and Exploratory Clinical Inves­ tigations of DEcision support systems driven by Artificial Intelligence),11 CLAIM (Checklist for Artificial Intelligence in Medical Imaging),12 and CONSORT-AI (Consolidated Standards of Reporting Trials-AI).13 In addition, Tam et al. introduced QUEST, a framework specifically for human eval­ uation of LLMs in healthcare.14 However, these guidelines do not cover RAG applications in clinical settings, emphasiz­ ing the need for a more specific guideline. Despite the promise of RAG in improving LLM performance in clinical settings, there is limited understanding of its overall effectiveness comparing with the baseline LLM, adoption in clinical domains, and optimal strategies for its development in biomedical applications. The aim of this study is to synthesize findings from recent research of RAG and LLM in biomedicine and provide clinical development guidelines to improve effec­ tiveness as well as transparency in future research. Materials and methods Study design We conducted a systematic literature review. The report was created in adherence to the Preferred Reporting Items for Sys­ tematic Reviews and Meta-Analyses 2020 analysis checklist (File S1).15 We also performed a meta-analysis following the Cochrane Handbook guidelines.16 Screening papers We searched in 3 databases (PubMed, Embase, and Psy­ cINFO), using terms related to “retrieval augmented gener­ ation” and “large language model.” PubMed and Embase were selected based on recommendations from the Cochrane Handbook, as they are widely recognized for their extensive coverage of biomedical literature.17 PsycINFO was included to capture articles from the psychological and behavioral sci­ ences. To maintain a high standard of quality and reliability, we focused on peer-reviewed articles and excluded preprints. The specific search terms used for each database are provided in File S2. Given that ChatGPT was released on November 30, 2022, we set the publication filter to search papers pub­ lished in 2023 and 2024. The search was performed on December 12, 2024. The inclusion criteria were: (1) the study must compare baseline LLM performance with RAG per­ formance and (2) the study must address a biomedical ques­ tion. The exclusion criteria were: (1) literature reviews, editorial comments, or viewpoint papers, (2) studies focusing on LLMs in languages other than English, or (3) studies cen­ tered on a multi-agent system without a focus on RAG. SL screened titles and abstracts, then conducted a full-text review of papers meeting the criteria. Data extraction For each included study, we extracted the following informa­ tion: author, title, publication year, journal, clinical task, and specialty. Regarding RAG techniques, we gathered details about the baseline LLM, retrieval sources, and strategies used in the pre-retrieval, retrieval, and post-retrieval stages. For evaluation, we extracted the evaluation method (human, automated, or a combination of both), the number of evalua­ tors, the evaluation dataset, and the evaluation metrics. Meta-analysis Effect size was defined as a metric quantifying the relation­ ship between variables, including both direction and magni­ tude.18 For each included study, we calculated the effect size between baseline LLM performance and RAG-enhanced LLM performance. The outcomes focused on the perform­ ance of generation results, such as accuracy and usefulness. Metrics related to the retrieval process, cost, or speed were not included as outcomes in the meta-analysis. For continu­ ous outcomes, we used Cohen’s d, standardized between- group mean difference (SMD), calculated as the difference in means divided by the pooled standard deviation. The stand­ ard error (SE) of SMD was calculated using the following for­ mula (1), where n1 and n2 represent the sample sizes of each group.19 For dichotomous measurements, we calculated the log-odds ratio, obtained by transforming the odds ratio (OR) with the natural logarithm, and the associated SE was calcu­ lated using formula (2), where a, b, c, and d represent the number of successful and failed events in the baseline LLM and RAG-enhanced LLM approaches. For studies reporting multiple outcomes, we used the overall outcome to calculate effect size. If no overall outcome was reported, we averaged the effect sizes of all reported outcomes. We excluded out­ comes with a sample size of less than 30 to avoid small- sample bias. SESMD ¼ ffiffiffiffiffiffiffiffiffiffiffiffiffiffiffiffiffiffiffiffiffiffiffiffiffiffiffiffiffiffiffiffiffiffiffiffiffiffiffiffiffiffiffiffiffiffiffiffiffiffi n1 þ n2 n1n2 þ SMD2 2 n1 þ n2 ð Þ s (1) SElogOR ¼ ffiffiffiffiffiffiffiffiffiffiffiffiffiffiffiffiffiffiffiffiffiffiffiffiffiffiffiffiffiffiffiffi 1 a þ 1 b þ 1 c þ 1 d r (2) We developed a random-effect meta-analysis model, because of the variability in RAG architectures and evalua­ tion datasets among the included studies. The random-effect model was used when individual study effects likely con­ tained additional sources of variance beyond sampling error. Between-study heterogeneity was assessed using Higgins & Thompson’s I2 statistic, where 25% indicated low heteroge­ neity, 50% moderate, and 75% substantial.20 We conducted subgroup analyses to explore performance variations across different factors. First, we analyzed the influence of the baseline LLM, referring to the foundation model (eg, GPT-4 or Llama2) that provides the core architec­ ture for the system. Second, we examined data retrieval strat­ egies, categorizing them as simple or complex. Simple strategies included fixed-length chunking and basic similarity search, and we performed a subgroup analysis to compare these with complex retrieval strategies. Third, we analyzed differences based on evaluation methods, distinguishing between human evaluations, such as Likert scale ratings for helpfulness and accuracy, and automatic evaluation metrics, including ROUGE-1 and BLEU. Finally, we conducted a sub­ group analysis based on the type of task, classifying studies 606 Journal of the American Medical Informatics Association, 2025, Vol. 32, No. 4 Downloaded from https://academic.oup.com/jamia/article/32/4/605/7954485 by guest on 11 January 2026
{ "author": "", "creationDate": "D:20250322110116+05'30'", "creationdate": "2025-03-22T11:01:16+05:30", "creator": "Servigistics Arbortext Advanced Print Publisher 11.1.4667/W", "file_path": "/home/donbr/open-biosciences/biosciences-research/data/raw/ocaf008.pdf", "format": "PDF 1.5", "keywords": "large language model; retrieval augmented generation; systematic review; meta-analysis", "modDate": "D:20260111011327+00'00'", "moddate": "2026-01-11T01:13:27+00:00", "page": 1, "producer": "PDFlib+PDI 9.0.7p3 (C++/Win32); modified using iTextSharp.LGPLv2.Core 3.7.4.0", "source": "/home/donbr/open-biosciences/biosciences-research/data/raw/ocaf008.pdf", "subject": "Doi: 10.1093/jamia/ocaf008 Journal of the American Medical Informatics Association, 32, 4, 2025 Publication Date: 15/01/2025 Abstract Objective The objectives of this study are to synthesize findings from recent research of retrieval-augmented generation (RAG) and large language models (LLMs) in biomedicine and provide clinical development guidelines to improve effectiveness.Materials and Methods We conducted a systematic literature review and a meta-analysis. The report was created in adherence to the Preferred Reporting Items for Systematic Reviews and Meta-Analyses 2020 analysis. Searches were performed in 3 databases (PubMed, Embase, PsycINFO) using terms related to “retrieval augmented generation” and “large language model,” for articles published in 2023 and 2024. We selected studies that compared baseline LLM performance with RAG performance. We developed a random-effect meta-analysis model, using odds ratio as the effect size.Results Among 335 studies, 20 were included in this literature review. The pooled effect size was 1.35, with a 95% confidence interval of 1.19-1.53, indicating a statistically significant effect (P = .001). We reported clinical tasks, baseline LLMs, retrieval sources and strategies, as well as evaluation methods.Discussion Building on our literature review, we developed Guidelines for Unified Implementation and Development of Enhanced LLM Applications with RAG in Clinical Settings to inform clinical applications using RAG.Conclusion Overall, RAG implementation showed a 1.35 odds ratio increase in performance compared to baseline LLMs. Future research should focus on (1) system-level enhancement: the combination of RAG and agent, (2) knowledge-level enhancement: deep integration of knowledge into LLM, and (3) integration-level enhancement: integrating RAG systems within electronic health records.", "title": "Improving large language model applications in biomedicine with retrieval-augmented generation: a systematic review, meta-analysis, and clinical development guidelines", "total_pages": 11, "trapped": "" }
into clinical decision-making and medical question- answering. These analyses provided insights into how varia­ tions in model architecture, retrieval strategies, evaluation methods, and task types affect system outcomes. To visualize the meta-analysis outcomes, we generated a forest plot. This plot displayed the effect size, confidence inter­ val for each study, as well as the pooled effect and predicted effect size. We evaluated the publication bias using a contour- enhanced funnel plot to investigate small-study effects. This scatter plot had the effect size on the x-axis and the inverted SE on the y-axis, with contours indicating P-values (<.1, .05, and .01).21 Symmetry in the funnel plot suggested no publica­ tion bias, and asymmetry was quantified using Egger’s regres­ sion test.22 We used the “meta” package in R to conduct the meta-analysis and perform statistical analyses. Results Study selection A total of 335 studies were identified from 3 databases: PubMed, Embase, and PsycINFO. After removing duplicates, 251 studies were screened. Of these, 20 studies were included in this literature review, all of which were published in 2024. One of the included studies was a conference paper.23 The flow diagram depicting the study selection process is shown in Figure 1. For each included study, their author, title, publi­ cation year, journal, clinical task, specialty, and retrieval sources are listed in Table S1 of File S2. Meta-analysis The pooled effect size was 1.35, with a 95% confidence inter­ val of 1.19-1.53, indicating a statistically significant effect (P ¼ .001). All outcomes and associated SEs are listed in File S2. The I2 value was 37%, indicating low to moderate heter­ ogeneity among the studies. The prediction interval ranged from 1.01 to 1.8. The forest plot is shown in Figure 2. The contour-enhanced funnel plot is presented in File S2. In Egger’s regression test, the intercept (β0) was 1.1, with a 95% confidence interval of [0.56, 1.64] and a P-value of .001, indicating the presence of small-study effects and potential publication bias. Figure 1. Flow diagram of included studies illustrating the systematic review process. Out of 335 records identified from PubMed, Embase, and PsycINFO, 84 duplicates were removed, leaving 251 records screened. After excluding 200 records, 51 reports were assessed for eligibility. 31 reports were excluded for reasons such as lack of RAG-LLM comparison, non-clinical focus, or commentary. Ultimately, 20 studies were included in the final review. LLM, large language model; RAG, retrieval-augmented generation. Journal of the American Medical Informatics Association, 2025, Vol. 32, No. 4 607 Downloaded from https://academic.oup.com/jamia/article/32/4/605/7954485 by guest on 11 January 2026
{ "author": "", "creationDate": "D:20250322110116+05'30'", "creationdate": "2025-03-22T11:01:16+05:30", "creator": "Servigistics Arbortext Advanced Print Publisher 11.1.4667/W", "file_path": "/home/donbr/open-biosciences/biosciences-research/data/raw/ocaf008.pdf", "format": "PDF 1.5", "keywords": "large language model; retrieval augmented generation; systematic review; meta-analysis", "modDate": "D:20260111011327+00'00'", "moddate": "2026-01-11T01:13:27+00:00", "page": 2, "producer": "PDFlib+PDI 9.0.7p3 (C++/Win32); modified using iTextSharp.LGPLv2.Core 3.7.4.0", "source": "/home/donbr/open-biosciences/biosciences-research/data/raw/ocaf008.pdf", "subject": "Doi: 10.1093/jamia/ocaf008 Journal of the American Medical Informatics Association, 32, 4, 2025 Publication Date: 15/01/2025 Abstract Objective The objectives of this study are to synthesize findings from recent research of retrieval-augmented generation (RAG) and large language models (LLMs) in biomedicine and provide clinical development guidelines to improve effectiveness.Materials and Methods We conducted a systematic literature review and a meta-analysis. The report was created in adherence to the Preferred Reporting Items for Systematic Reviews and Meta-Analyses 2020 analysis. Searches were performed in 3 databases (PubMed, Embase, PsycINFO) using terms related to “retrieval augmented generation” and “large language model,” for articles published in 2023 and 2024. We selected studies that compared baseline LLM performance with RAG performance. We developed a random-effect meta-analysis model, using odds ratio as the effect size.Results Among 335 studies, 20 were included in this literature review. The pooled effect size was 1.35, with a 95% confidence interval of 1.19-1.53, indicating a statistically significant effect (P = .001). We reported clinical tasks, baseline LLMs, retrieval sources and strategies, as well as evaluation methods.Discussion Building on our literature review, we developed Guidelines for Unified Implementation and Development of Enhanced LLM Applications with RAG in Clinical Settings to inform clinical applications using RAG.Conclusion Overall, RAG implementation showed a 1.35 odds ratio increase in performance compared to baseline LLMs. Future research should focus on (1) system-level enhancement: the combination of RAG and agent, (2) knowledge-level enhancement: deep integration of knowledge into LLM, and (3) integration-level enhancement: integrating RAG systems within electronic health records.", "title": "Improving large language model applications in biomedicine with retrieval-augmented generation: a systematic review, meta-analysis, and clinical development guidelines", "total_pages": 11, "trapped": "" }
Clinical applications of RAG RAG techniques have been applied across a broad range of medical specialties, as shown in Table 1. These applications include clinical decision-making and medical question- answering. In clinical decision making, RAG has supported personalized treatment,23,24 emergency triage,25 and disease management.26,27 For medical question-answering, RAG’s capability has been explored to address complex treatment guidelines questions,28 as well as queries focused on specific areas, such as head and neck surgery-related questions,29 and patient questions regarding diabetes.30 In the subgroup anal­ ysis, 13 studies focused on clinical decision-making (OR 1.46, 95% CI [1.16, 1.71]) and 7 studies focused on medical question-answering (OR 1.32, 95% CI [1.08, 1.63]), with no statistically significant difference observed between these 2 groups. Baseline LLMs The baseline LLMs varied across studies, with GPT-4 being the most common, used in 14 studies, (OR: 1.58, 95% CI: 1.21-2.04). GPT-3.5, used in 6 studies, showed an OR of 1.43 (95% CI: 1.06-1.93). Llama2 was applied in 5 studies (OR: 1.25, 95% CI: 1.08-1.44). Retrieval sources Retrieval sources were categorized as pre-stored documents and real-time online browsing. Regarding pre-stored docu­ ments, 6 studies used clinical guidelines, such as the Emer­ gency Severity Index (ESI) Ver.3 Field Triage.25 Five studies used academic articles from sources like PubMed abstracts or full texts, or document sets such as the Radiographics Top 10 Reading List on Gastrointestinal Imaging.31 Three studies used specialized knowledge bases, including ChatENT, OncoKB, and RareDis Corpus, while one study employed a general biomedical knowledge graph (Scalable Precision Medicine Open Knowledge Engine [SPOKE]). SPOKE Table 1. Distribution of RAG applications by medical specialty and task type. Medical Specialty Frequency Internal medicine 4 General medicine 3 Oncology 3 Emergency medicine 2 Gastroenterology 2 Otolaryngology 1 Hepatology 1 Rare diseases 1 Orthopedics 1 Neurology 1 Ophthalmology 1 Task Frequency (%) Clinical decision-making 13 (65%) Medical question-answering 7 (35%) Figure 2. A forest plot showing the odds ratio (OR) of total impacts of the RAG-enhanced system compared with the baseline LLM system in clinical tasks. The left panel lists individual studies (e.g., Zakka et al., Murugan et al., Long et al.) along with their corresponding ORs and 95% confidence intervals (CIs). The right panel visualizes these ORs, with square markers proportional to study weights and horizontal lines representing CIs. The overall pooled OR is 1.35 (95% CI: 1.19–1.53), represented by a diamond at the bottom. 608 Journal of the American Medical Informatics Association, 2025, Vol. 32, No. 4 Downloaded from https://academic.oup.com/jamia/article/32/4/605/7954485 by guest on 11 January 2026
{ "author": "", "creationDate": "D:20250322110116+05'30'", "creationdate": "2025-03-22T11:01:16+05:30", "creator": "Servigistics Arbortext Advanced Print Publisher 11.1.4667/W", "file_path": "/home/donbr/open-biosciences/biosciences-research/data/raw/ocaf008.pdf", "format": "PDF 1.5", "keywords": "large language model; retrieval augmented generation; systematic review; meta-analysis", "modDate": "D:20260111011327+00'00'", "moddate": "2026-01-11T01:13:27+00:00", "page": 3, "producer": "PDFlib+PDI 9.0.7p3 (C++/Win32); modified using iTextSharp.LGPLv2.Core 3.7.4.0", "source": "/home/donbr/open-biosciences/biosciences-research/data/raw/ocaf008.pdf", "subject": "Doi: 10.1093/jamia/ocaf008 Journal of the American Medical Informatics Association, 32, 4, 2025 Publication Date: 15/01/2025 Abstract Objective The objectives of this study are to synthesize findings from recent research of retrieval-augmented generation (RAG) and large language models (LLMs) in biomedicine and provide clinical development guidelines to improve effectiveness.Materials and Methods We conducted a systematic literature review and a meta-analysis. The report was created in adherence to the Preferred Reporting Items for Systematic Reviews and Meta-Analyses 2020 analysis. Searches were performed in 3 databases (PubMed, Embase, PsycINFO) using terms related to “retrieval augmented generation” and “large language model,” for articles published in 2023 and 2024. We selected studies that compared baseline LLM performance with RAG performance. We developed a random-effect meta-analysis model, using odds ratio as the effect size.Results Among 335 studies, 20 were included in this literature review. The pooled effect size was 1.35, with a 95% confidence interval of 1.19-1.53, indicating a statistically significant effect (P = .001). We reported clinical tasks, baseline LLMs, retrieval sources and strategies, as well as evaluation methods.Discussion Building on our literature review, we developed Guidelines for Unified Implementation and Development of Enhanced LLM Applications with RAG in Clinical Settings to inform clinical applications using RAG.Conclusion Overall, RAG implementation showed a 1.35 odds ratio increase in performance compared to baseline LLMs. Future research should focus on (1) system-level enhancement: the combination of RAG and agent, (2) knowledge-level enhancement: deep integration of knowledge into LLM, and (3) integration-level enhancement: integrating RAG systems within electronic health records.", "title": "Improving large language model applications in biomedicine with retrieval-augmented generation: a systematic review, meta-analysis, and clinical development guidelines", "total_pages": 11, "trapped": "" }
integrates over 40 publicly available biomedical knowledge sources across separate domains, such as genes, proteins, drugs, compounds, and diseases, along with their known relationships.32 Two studies used textbooks, such as Harri­ son's Principles of Internal Medicine, while 3 others utilized electronic health record (EHR) data. Additionally, Zakka et al. added over 500 markdown files from MDCalc to improve clinical calculation capabilities in LLM.28 Two stud­ ies employed real-time online browsing to search academic sites, such as PubMed and UpToDate. The amount of retrieval resources varied across studies, ranging from a small dataset specific to 6 osteoarthritis guidelines to a large dataset of EHR data from 7 hospitals. Retrieval strategies Identified retrieval strategies were grouped based on the RAG stages: pre-retrieval, retrieval, and post-retrieval. Figure 3 presents an example of how RAG is applied and lists identi­ fied strategies within each stage. In the pre-retrieval stage, 50% of studies (n ¼ 10) reported strategies, such as query rewriting, document preprocessing, and assessing the necessity of retrieval. Zakka et al. simplified queries by rephrasing text into search terms that are better suited for website browsing,28 while Wang et al. focused on techniques such as correcting errors, expanding abbrevia­ tions, and matching synonyms in user queries.30 Soman et al. extracted disease entities in queries and retrieved correspond­ ing nodes from a knowledge graph.33 Document preprocess­ ing involved removing non-textual elements from PMC papers (eg, figures, references, and author disclosures),30 extracted tables from PDFs using pdfplumber, structured the content with pydantic for seamless integration.25 In addition to query modification and document preprocessing, Jeong et al. fine-tuned a model to determine whether retrieval was necessary for a given query.34 During the data retrieval stage, 85% of studies (n ¼ 17) reported strategies regarding indexing, aligning queries with documents, and ranking retrieval chunks. Chunking methods ranged from fixed-size chunks35 to recursive splits.36 Embed­ ding models such as Text-embedding-ada-002,24,28–30,36,37 MiniLM, and PubMedBERT33 were commonly used to con­ vert sentences into vectors. Cosine similarity was the primary metric for measuring query-document alignment. Two stud­ ies adopted Maximal Marginal Relevance for search and highlighted its improved performance over similarity-based methods.24,35 A domain-specific retriever, MedCPT, was used in one study.34 Another study used the multi-vector retriever that leveraged summarized document sections to identify the original content for final answer generation.25 The retrieval cutoff parameters varied widely, with probabil­ ity thresholds up to 0.83 and the number of retrieved chunks ranging from 3 to 90.28,36,38 Vector databases like FAISS and Chroma were frequently reported, and LangChain was widely used for document processing and retrieval.23,25,35,38 In the subgroup analysis, 12 studies used simple data retrieval strategies (OR 1.30, 95% CI [1.16, 1.45]), while 5 studies used complex data retrieval strategies (OR 1.30, 95% CI [1.07, 1.24]), with no statistically significant difference observed between the 2 approaches. In the post-retrieval stage, 65% of studies (n ¼ 13) imple­ mented specific strategies to refine outputs. Murugan et al. tailored prompts by providing clear context, defining roles (eg, distinguishing between healthcare providers and patients to deliver appropriately detailed information), and incorpo­ rating relevant citations from retrieval sources such as the Clinical Pharmacogenetics Implementation Consortium guidelines and Food and Drug Administration (FDA) label­ ing.24 Soman et al. utilized prompt engineering to integrate accurate knowledge sources and statistical evidence, such as P-values and z-scores, from the SPOKE knowledge graph into their outputs.33 Wang et al. outlined a detailed process in the post-retrieval stage using prompt engineering, which involved decomposing retrieved text into individual claims, verifying each claim with external knowledge sources, Figure 3. An example of using RAG in clinical applications, with identified strategies in 3 stages: pre-retrieval, retrieval, and post-retrieval. The user query seeks recommended medications for symptomatic peripheral artery disease without increased bleeding risk. The system retrieves evidence from clinical guidelines, processes it through embeddings and a vector database, and outputs a response, including rivaroxaban with low-dose aspirin, as recommended by the retrieved guideline information. In this example, GPT-4 suggested a dose of 75 mg Aspirin, but the common low-dose Aspirin is 81 mg. Journal of the American Medical Informatics Association, 2025, Vol. 32, No. 4 609 Downloaded from https://academic.oup.com/jamia/article/32/4/605/7954485 by guest on 11 January 2026
{ "author": "", "creationDate": "D:20250322110116+05'30'", "creationdate": "2025-03-22T11:01:16+05:30", "creator": "Servigistics Arbortext Advanced Print Publisher 11.1.4667/W", "file_path": "/home/donbr/open-biosciences/biosciences-research/data/raw/ocaf008.pdf", "format": "PDF 1.5", "keywords": "large language model; retrieval augmented generation; systematic review; meta-analysis", "modDate": "D:20260111011327+00'00'", "moddate": "2026-01-11T01:13:27+00:00", "page": 4, "producer": "PDFlib+PDI 9.0.7p3 (C++/Win32); modified using iTextSharp.LGPLv2.Core 3.7.4.0", "source": "/home/donbr/open-biosciences/biosciences-research/data/raw/ocaf008.pdf", "subject": "Doi: 10.1093/jamia/ocaf008 Journal of the American Medical Informatics Association, 32, 4, 2025 Publication Date: 15/01/2025 Abstract Objective The objectives of this study are to synthesize findings from recent research of retrieval-augmented generation (RAG) and large language models (LLMs) in biomedicine and provide clinical development guidelines to improve effectiveness.Materials and Methods We conducted a systematic literature review and a meta-analysis. The report was created in adherence to the Preferred Reporting Items for Systematic Reviews and Meta-Analyses 2020 analysis. Searches were performed in 3 databases (PubMed, Embase, PsycINFO) using terms related to “retrieval augmented generation” and “large language model,” for articles published in 2023 and 2024. We selected studies that compared baseline LLM performance with RAG performance. We developed a random-effect meta-analysis model, using odds ratio as the effect size.Results Among 335 studies, 20 were included in this literature review. The pooled effect size was 1.35, with a 95% confidence interval of 1.19-1.53, indicating a statistically significant effect (P = .001). We reported clinical tasks, baseline LLMs, retrieval sources and strategies, as well as evaluation methods.Discussion Building on our literature review, we developed Guidelines for Unified Implementation and Development of Enhanced LLM Applications with RAG in Clinical Settings to inform clinical applications using RAG.Conclusion Overall, RAG implementation showed a 1.35 odds ratio increase in performance compared to baseline LLMs. Future research should focus on (1) system-level enhancement: the combination of RAG and agent, (2) knowledge-level enhancement: deep integration of knowledge into LLM, and (3) integration-level enhancement: integrating RAG systems within electronic health records.", "title": "Improving large language model applications in biomedicine with retrieval-augmented generation: a systematic review, meta-analysis, and clinical development guidelines", "total_pages": 11, "trapped": "" }
conducting safety checks by applying 24 predefined rules to ensure ethical and factual accuracy, and summarizing the results.30 Glicksberg et al. developed an ensemble model that combined structured and unstructured data to predict hospi­ tal admission probabilities. These predicted probabilities, along with similar historical cases, were incorporated into the prompt to enhance the performance of LLM.37 Chen et al. used Chain-of-Thought (CoT) prompting to improve LLM reasoning capabilities.39 Kresevic et al. customized prompts to help the model interpret structured guidelines, combined with few-shot learning using 54 question-answer pairs.27 Jeong et al. fine-tuned LLMs to assess the relevance of retrieved evidence, ensure all statements were evidence-based, and confirm that the response effectively addressed the query.34 Evaluation Nine studies used human evaluation, 8 relied on automated evaluation (eg, similarity comparisons between generated sentences and original answers), and 3 used a mix of both. Outcomes from human evaluation showed an overall OR of 1.65 (95% CI: 1.36-2.03), while automatic evaluation resulted in an OR of 1.20 (95% CI: 1.1-1.41). The differen­ ces between the 2 were statistically significant (P < .01). There were 4 human evaluators on average, with the range spanning from 1 to 10. Most human evaluators were physicians from relevant specialties according to the study focus. In one case, 3 diabetic patients were involved in evaluating the understandability of diabetes-related patient queries.30 Twelve studies used self-curated datasets focused on research tasks. Examples included the ClinicalQA bench­ mark, which comprised 314 open-ended questions about treatment guidelines and clinical calculations generated by physicians,28 and 43 diabetes-related questions sourced from the National Institute of Diabetes and Digestive and Kidney Diseases website.30 Simulated cases from medical examina­ tions were also utilized.25 Three studies used EHR data.35,37,40 Six studies used public benchmark datasets, such US board exam practice questions, MedMCQA29,34 and longform question-answering benchmarks (eg, LiveQA, Med­ icationQA).34 The self-curated datasets averaged 76 ques­ tions, ranging from 7 to 314. The length of public benchmark datasets varied significantly, from 102 questions in the LiveQA dataset28 to 194 000 questions in the MedMCQA dataset.34 Most studies reported evaluation metrics for the final response generation, while 4 (25%) also included specific metrics to evaluate the retrieval process. For instance, 1 study measured recall in context retrieval,24 another evaluated retrieval accuracy,33 and a fine-tuned LLM was developed to assess the relevance of retrieved information to the user’s query.34 Additionally, 1 study evaluated the accuracy of using LLMs to extract text from figures and tables during document preprocessing.27 The final evaluation metrics focused on the generated responses, consistent with those used in LLM-only systems. These metrics could be catego­ rized as accuracy, completeness, user perception, safety, hal­ lucination, citation, bias, and language. Accuracy was the most frequently reported metric, covering Likert scale rat­ ings, match rates, correct treatment percentages,9 AUC, AUPRC, and F1 scores, as well as text similarity metrics like ROUGE (ROUGE-1, ROUGE-2, ROUGE-L), BLEU, METEOR, and BERTScore,21 which compared LLM- generated responses to expert-provided answers. Complete­ ness metrics assessed whether responses included all neces­ sary information, typically using Likert scales. User perception captured subjective feedback from both healthcare providers and patients on understandability, helpfulness, and whether responses met user intent, usually using Likert scales. Safety metrics focused both on user-related and system- related aspects. These metrics assessed potential harm, adver­ sarial safety, and risk management,24 ensuring that outputs were free of harmful content or risks. Scientific validity and adherence to evidence were also evaluated.29 One study used adversarial prompting, defined as intentionally adding harm­ ful directives to a prompt, to evaluate the safety of the RAG system.28 Hallucinations were primarily identified through manual review, with definitions varying across studies. Some studies defined hallucinations as nonfactual information, while one study added 2 other types of hallucinations: input- conflicting (content deviating from user-provided input) and contextual-conflicting (content conflicting with previously generated information).27,41 Citation metrics measured the accuracy of provided references, with valid references consid­ ered those that pointed to established publications, guide­ lines, or research. Bias and language were evaluated for clarity and neutrality, ensuring responses were unbiased and empathetic to patient concerns.24 Discussion This study presents a systematic review of current research on RAG for clinical tasks. Overall, RAG implementation increased outcomes by 1.35 times compared to baseline LLM. We analyzed clinical tasks, baseline LLMs, retrieval sources and strategies, as well as evaluation methods. Despite the potential benefits of RAG systems, there remains room for improvement. Building on our literature review, we devel­ oped GUIDE-RAG (Guidelines for Unified Implementation and Development of Enhanced LLM Applications with RAG in Clinical Settings) for future clinical applications using RAG (Figure 4). GUIDE-RAG: 1) Define clear clinical tasks and evaluation datasets. Future research should clearly define clinical tasks and questions to maximize the effectiveness of RAGs. Ambi­ guity in questions can hinder performance, particularly in less powerful LLMs, making it challenging to achieve significant improvements in responses generation, even with improved knowledge selection.42 For example, one study in the review constructed the evaluation dataset using 30 case reports on rare diseases from PubMed.38 The authors did not report human performance on the self-build dataset. The questions themselves might have been inherently challenging or ambiguous. As expected, the reported performance showed modest improvement, with an OR of 1.31. 2) Identify appropriate external resources for specific clini­ cal tasks. The first step in developing a RAG-based clinical system is to identify external resources that fill the knowledge gaps of the baseline LLM in relation to specific clinical tasks. The external knowledge should complement the LLM’s existing capabilities to effectively address task 610 Journal of the American Medical Informatics Association, 2025, Vol. 32, No. 4 Downloaded from https://academic.oup.com/jamia/article/32/4/605/7954485 by guest on 11 January 2026
{ "author": "", "creationDate": "D:20250322110116+05'30'", "creationdate": "2025-03-22T11:01:16+05:30", "creator": "Servigistics Arbortext Advanced Print Publisher 11.1.4667/W", "file_path": "/home/donbr/open-biosciences/biosciences-research/data/raw/ocaf008.pdf", "format": "PDF 1.5", "keywords": "large language model; retrieval augmented generation; systematic review; meta-analysis", "modDate": "D:20260111011327+00'00'", "moddate": "2026-01-11T01:13:27+00:00", "page": 5, "producer": "PDFlib+PDI 9.0.7p3 (C++/Win32); modified using iTextSharp.LGPLv2.Core 3.7.4.0", "source": "/home/donbr/open-biosciences/biosciences-research/data/raw/ocaf008.pdf", "subject": "Doi: 10.1093/jamia/ocaf008 Journal of the American Medical Informatics Association, 32, 4, 2025 Publication Date: 15/01/2025 Abstract Objective The objectives of this study are to synthesize findings from recent research of retrieval-augmented generation (RAG) and large language models (LLMs) in biomedicine and provide clinical development guidelines to improve effectiveness.Materials and Methods We conducted a systematic literature review and a meta-analysis. The report was created in adherence to the Preferred Reporting Items for Systematic Reviews and Meta-Analyses 2020 analysis. Searches were performed in 3 databases (PubMed, Embase, PsycINFO) using terms related to “retrieval augmented generation” and “large language model,” for articles published in 2023 and 2024. We selected studies that compared baseline LLM performance with RAG performance. We developed a random-effect meta-analysis model, using odds ratio as the effect size.Results Among 335 studies, 20 were included in this literature review. The pooled effect size was 1.35, with a 95% confidence interval of 1.19-1.53, indicating a statistically significant effect (P = .001). We reported clinical tasks, baseline LLMs, retrieval sources and strategies, as well as evaluation methods.Discussion Building on our literature review, we developed Guidelines for Unified Implementation and Development of Enhanced LLM Applications with RAG in Clinical Settings to inform clinical applications using RAG.Conclusion Overall, RAG implementation showed a 1.35 odds ratio increase in performance compared to baseline LLMs. Future research should focus on (1) system-level enhancement: the combination of RAG and agent, (2) knowledge-level enhancement: deep integration of knowledge into LLM, and (3) integration-level enhancement: integrating RAG systems within electronic health records.", "title": "Improving large language model applications in biomedicine with retrieval-augmented generation: a systematic review, meta-analysis, and clinical development guidelines", "total_pages": 11, "trapped": "" }
requirements. For instance, in question-answering tasks related to broad medical exams for physicians, clinical guidelines (eg, StatPearls) and textbooks proved more useful than PubMed abstracts as external sources.43 Another example from our review involved a task focused on medical question-answering in internal medi­ cine. The study used a single source—Harrison’s Princi­ ples of Internal Medicine—as the knowledge retrieval source, and the reported improvement was marginal (OR: 1.14).36 Expanding the knowledge base to include additional resources, such as clinical guidelines, could potentially enhance the performance of the RAG system for such tasks. 3) Preprocess multi-modal content in documents. Clinical guidelines and medical literature often contain complex information presented through flowcharts, graphs, and tables. Accurately parsing this multi-modal content is essential for effective retrieval. Relying solely on LLMs for text extraction may be insufficient; a pre­ liminary study found that GPT-4 Turbo had only a 16% accuracy rate in extracting table data.27 Compre­ hensive document preprocessing should systematically extract relevant information from text, tables, and fig­ ures to ensure accuracy and clarity. Only 3 studies in our review explicitly mentioned extracting text from tables or figures during the pre-retrieval process.25,27,44 4) Optimize chunking and indexing based corpus structure. The structure of clinical knowledge corpora should be carefully considered during chunking and indexing. Fixed-length chunking can introduce noise by fragment­ ing related information, which can reduce retrieval accuracy. Researchers should optimize the chunking granularity based on a thorough review of the clinical knowledge corpus, to ensure the completeness of retrieved information. An alternative approach is dynamic chunking, which adjusts chunk boundaries based on semantic similarity changes.45 Other approaches include recursive chunking, which hierarchi­ cally divides text into smaller chunks using delimiters like headings, subheadings, paragraphs, and sentences. Sliding window chunking enables layered retrieval by overlapping chunks of text, allowing the system to cap­ ture and merge contextually related information across different segments.46 Context enriched chucking enhan­ ces retrieval by incorporating concise summaries within each segment to provide additional context for down­ stream tasks.47 In indexing, while dense indexing (con­ verting text to vectors) is widely used, it may miss global information. The structure of a clinical knowledge cor­ pora such as some headings, keywords, can be used as sparse indexing and further combined with dense index­ ing. This hybrid approach that combines dense and sparse indexing can improve retrieval performance by capturing both global and local information.48,49 5) Explore advanced retrieval strategies, including iterative and graph-based retrieval. Iterative retrieval improves accuracy by refining results through multiple rounds. Parameters such as the num­ ber of retrieved chunks or cutoff thresholds should be optimized based on specific clinical questions, as retrieval needs can vary—some questions may not require external knowledge at all. Researchers should evaluate retrieval requirements in advance and adapt retrieval parameters accordingly. Graph-based retrieval, which structures entities and relationships into a graph, can improve information synthesis from multiple sour­ ces. For example, GraphRAG identified entities and relationships from documents and developed a graph using LLM. Then, they used clustering algorithm to Figure 4. Overview of GUIDE-RAG: This framework streamlines RAG in clinical applications through three iterative stages. In the pre-retrieval stage, it focuses on defining tasks, identifying relevant resources, and preprocessing content. The retrieval stage enhances data retrieval with optimized chunking, indexing, and advanced strategies such as graph-based retrieval. The post-retrieval stage emphasizes system evaluation, monitoring, knowledge updates, and implementing few-shot learning for complex clinical reasoning, ensuring robust and adaptive performance. CoT, chain-of-thought. Journal of the American Medical Informatics Association, 2025, Vol. 32, No. 4 611 Downloaded from https://academic.oup.com/jamia/article/32/4/605/7954485 by guest on 11 January 2026
{ "author": "", "creationDate": "D:20250322110116+05'30'", "creationdate": "2025-03-22T11:01:16+05:30", "creator": "Servigistics Arbortext Advanced Print Publisher 11.1.4667/W", "file_path": "/home/donbr/open-biosciences/biosciences-research/data/raw/ocaf008.pdf", "format": "PDF 1.5", "keywords": "large language model; retrieval augmented generation; systematic review; meta-analysis", "modDate": "D:20260111011327+00'00'", "moddate": "2026-01-11T01:13:27+00:00", "page": 6, "producer": "PDFlib+PDI 9.0.7p3 (C++/Win32); modified using iTextSharp.LGPLv2.Core 3.7.4.0", "source": "/home/donbr/open-biosciences/biosciences-research/data/raw/ocaf008.pdf", "subject": "Doi: 10.1093/jamia/ocaf008 Journal of the American Medical Informatics Association, 32, 4, 2025 Publication Date: 15/01/2025 Abstract Objective The objectives of this study are to synthesize findings from recent research of retrieval-augmented generation (RAG) and large language models (LLMs) in biomedicine and provide clinical development guidelines to improve effectiveness.Materials and Methods We conducted a systematic literature review and a meta-analysis. The report was created in adherence to the Preferred Reporting Items for Systematic Reviews and Meta-Analyses 2020 analysis. Searches were performed in 3 databases (PubMed, Embase, PsycINFO) using terms related to “retrieval augmented generation” and “large language model,” for articles published in 2023 and 2024. We selected studies that compared baseline LLM performance with RAG performance. We developed a random-effect meta-analysis model, using odds ratio as the effect size.Results Among 335 studies, 20 were included in this literature review. The pooled effect size was 1.35, with a 95% confidence interval of 1.19-1.53, indicating a statistically significant effect (P = .001). We reported clinical tasks, baseline LLMs, retrieval sources and strategies, as well as evaluation methods.Discussion Building on our literature review, we developed Guidelines for Unified Implementation and Development of Enhanced LLM Applications with RAG in Clinical Settings to inform clinical applications using RAG.Conclusion Overall, RAG implementation showed a 1.35 odds ratio increase in performance compared to baseline LLMs. Future research should focus on (1) system-level enhancement: the combination of RAG and agent, (2) knowledge-level enhancement: deep integration of knowledge into LLM, and (3) integration-level enhancement: integrating RAG systems within electronic health records.", "title": "Improving large language model applications in biomedicine with retrieval-augmented generation: a systematic review, meta-analysis, and clinical development guidelines", "total_pages": 11, "trapped": "" }
offer global information based on user query, offering better performance than naïve RAG on the traditional vector databases.50 6) Implement few-shot learning with CoT for complex clin­ ical reasoning. Few-shot learning has been shown to enhance LLMs’ reasoning capabilities by teaching specific reasoning that may not have been included in their original training. Similarly, CoT techniques can improve complex reason­ ing in clinical tasks.51,52 Researchers should generate high-quality examples and incorporate CoT strategies into the final query to refine specialized reasoning. 7) Evaluate all stages of the RAG system development. Most current studies focus only on final performance, overlooking the importance of evaluating each stage of development. It is crucial to formally assess and report performance at the pre-retrieval, retrieval, and post- retrieval stages. Evaluating the knowledge boundaries of the baseline LLM, potential conflicts between the LLM and external knowledge, and the accuracy and coverage of retrieved information helps ensure replicability and transparency. This level of evaluation enables other researchers to understand why a RAG system works (or does not) and facilitates reproducibility. 8) Consider cost and time constraints. Advanced retrieval strategies can improve performance but often increase processing time and computational costs. For example, graph-based RAG requires substan­ tial resources for developing knowledge graphs, and responses from global summaries may take longer than with naïve RAG methods.50 Another example is to fine- tune LLMs to evaluate the needs and the quality of retrieval.34 In terms of computational cost, this process is expensive, especially when scaling the method to larger datasets or deploying it in a real-time system. Also, a set of extra processes will make the whole speed slow. The long response time might have a nonignorable negative impact in situations that need a quick answer, especially common in clinical settings. Researchers should balance performance improvements with time and cost considerations. Only 1 study in our review, which focused on gastrointestinal radiology diagnosis based on imaging descriptions, compared the cost and response time between LLMs and LLMs with RAG.31 The mean response time was 29.8 s for LLM with RAG vs 15.7s for LLM alone, with costs of $0.15 and $0.02 per case, respectively. Another study used EHR to pre­ dict cognitive decline only reported cost, with LLM: $4.49; RAG: $12.51. Another study that used EHR data to predict cognitive decline reported costs of $4.49 for LLM alone and $12.51 for LLM with RAG.53 9) Establish monitoring and knowledge update strategies. An important concept in AI applications in healthcare, algorithmovigilance, which defined as “scientific meth­ ods and activities relating to the evaluation, monitoring, understanding, and prevention of adverse effects of algorithms in health care,” 54 should also be considered in the RAG applications. Researchers need to develop long-term monitoring strategies for the RAG system per­ formance, especially in clinical applications. In addition, current studies use fixed external datasets. Researchers should update external knowledge sources as latest information becomes available. Clear strategies for updating knowledge should be defined, specifying when and how updates will occur. For future studies, the first direction could be the system- level enhancement, the combination of RAG and LLM- powered agents. LLM-powered agents are AI systems that use LLMs with complex reasoning and planning capabilities, memory management, interactive capabilities with the envi­ ronment, and actions to execute tasks.55,56 Recent research points to the emerging trend of combination of RAG and LLM-powered agents, where agents can assist in planning and decision making for complex tasks, rather than simple retrieval.57 For example, clinicians and patients have diverse information access needs, some needing to analyze text from a knowledge base, others needing to incorporate structured data from an EHR. RAG will eventually only become one of the methods for agents to access information. Moreover, future research could focus on the usage of internal and external functions and tools, long-term and short-term mem­ ory module, self-learning module. For example, a study developed an agent to answer questions related to rare dis­ eases by expanding beyond RAG with additional tool func­ tions, such as querying phenotypes and performing web searches. This approach improved the overall correctness from 0.48 to 0.75 compared to the GPT-4 baseline LLM.58 The second future direction could focus on the knowledge- level enhancement: deep integration of external knowledge into LLM. LLM exhibits the knowledge boundaries. RAG approaches retrieving external knowledge and then integrates it into LLMs in the forms of prompts for the final generation to enhance the capabilities of LLMs in perceiving knowledge boundaries.59 However, the integration of external knowl­ edge into LLM reasoning is typically limited to providing the retrieved data as additional context for the LLM’s query dur­ ing generation. This approach keeps retrieval and generation loosely connected, and the LLM’s output can still be influ­ enced by its inherent knowledge boundaries or by noise in the retrieved text, leading to incorrect answers. Additionally, when the external knowledge source is EHR data, this enhancement becomes even more important. Current EHR data is organized in a “problem-oriented medical record” (POMR) format, which collects and displays information in a structured manner.60 LLMs excel in free-form contexts, and their ability to perform clinical tasks depends on access to unstructured text that provides a comprehensive view of the patient. Achieving this within the structured POMR format in modern EHR systems poses a significant challenge.61 Therefore, investigating how to realize the deep integration of external knowledge with LLM reasoning is an important direction for future research in clinical applications. The final direction is the integration-level enhancement, focusing on integrating RAG systems within EHRs. Current research has primarily focused on development and testing outside of EHR systems. To seamlessly provide support for healthcare providers and patients, future efforts should pri­ oritize embedding RAG systems into EHR interfaces. This requires collaboration with EHR vendors to ensure the neces­ sary infrastructure is available. Researchers also can facilitate this integration using data exchange frameworks, such as SMART on FHIR.62 612 Journal of the American Medical Informatics Association, 2025, Vol. 32, No. 4 Downloaded from https://academic.oup.com/jamia/article/32/4/605/7954485 by guest on 11 January 2026
{ "author": "", "creationDate": "D:20250322110116+05'30'", "creationdate": "2025-03-22T11:01:16+05:30", "creator": "Servigistics Arbortext Advanced Print Publisher 11.1.4667/W", "file_path": "/home/donbr/open-biosciences/biosciences-research/data/raw/ocaf008.pdf", "format": "PDF 1.5", "keywords": "large language model; retrieval augmented generation; systematic review; meta-analysis", "modDate": "D:20260111011327+00'00'", "moddate": "2026-01-11T01:13:27+00:00", "page": 7, "producer": "PDFlib+PDI 9.0.7p3 (C++/Win32); modified using iTextSharp.LGPLv2.Core 3.7.4.0", "source": "/home/donbr/open-biosciences/biosciences-research/data/raw/ocaf008.pdf", "subject": "Doi: 10.1093/jamia/ocaf008 Journal of the American Medical Informatics Association, 32, 4, 2025 Publication Date: 15/01/2025 Abstract Objective The objectives of this study are to synthesize findings from recent research of retrieval-augmented generation (RAG) and large language models (LLMs) in biomedicine and provide clinical development guidelines to improve effectiveness.Materials and Methods We conducted a systematic literature review and a meta-analysis. The report was created in adherence to the Preferred Reporting Items for Systematic Reviews and Meta-Analyses 2020 analysis. Searches were performed in 3 databases (PubMed, Embase, PsycINFO) using terms related to “retrieval augmented generation” and “large language model,” for articles published in 2023 and 2024. We selected studies that compared baseline LLM performance with RAG performance. We developed a random-effect meta-analysis model, using odds ratio as the effect size.Results Among 335 studies, 20 were included in this literature review. The pooled effect size was 1.35, with a 95% confidence interval of 1.19-1.53, indicating a statistically significant effect (P = .001). We reported clinical tasks, baseline LLMs, retrieval sources and strategies, as well as evaluation methods.Discussion Building on our literature review, we developed Guidelines for Unified Implementation and Development of Enhanced LLM Applications with RAG in Clinical Settings to inform clinical applications using RAG.Conclusion Overall, RAG implementation showed a 1.35 odds ratio increase in performance compared to baseline LLMs. Future research should focus on (1) system-level enhancement: the combination of RAG and agent, (2) knowledge-level enhancement: deep integration of knowledge into LLM, and (3) integration-level enhancement: integrating RAG systems within electronic health records.", "title": "Improving large language model applications in biomedicine with retrieval-augmented generation: a systematic review, meta-analysis, and clinical development guidelines", "total_pages": 11, "trapped": "" }
Limitations This study was limited to peer-reviewed publications avail­ able in biomedical databases (eg, PubMed, Embase), exclud­ ing preprint articles from repositories like ArXiv. Additionally, only studies in English language were included, which might have excluded relevant studies in other lan­ guages. We did not include sources such as IEEE Xplore or Google Scholar, which might have additional relevant stud­ ies. However, our focus was on biomedicine, and we priori­ tized databases specifically tailored to biomedical research to maintain the relevance and quality of the included studies. Furthermore, we used free-text searches in the databases, which activated automatic mapping to Medical Subject Headings (MeSH) and Emtree terms, improving retrieval accuracy. However, the limitations of automatic term map­ ping cannot be ignored, as it may introduce variability if the underlying algorithms change. To address this, we have docu­ mented all identified papers from our search. The title, publi­ cation year, PMID, PUI, and database source for each study are provided in File S3. Conclusion We conducted a systematic literature review of studies exploring the use of RAG and LLM in clinical tasks. RAG implementation showed a 1.35 odds ratio increase in per­ formance compared to baseline LLMs. To improve perform­ ance and transparency in future studies, we developed guidelines for improving clinical RAG applications based on current research findings. Future research could focus on these 3 directions: (1) system-level enhancement: the combi­ nation of RAG and agent, (2) knowledge-level enhancements: deep integration of knowledge into LLM, and (3) integration- level enhancements: integrating RAG systems within EHRs. Author contributions Siru Liu (Conceptualization, Data curation, Formal analysis, Investigation, Methodology, Software, Visualization, Writing – original draft), Allison B. McCoy (Conceptualization, Writ­ ing – review & editing), Adam Wright (Conceptualization, Writing – review & editing). Supplementary material Supplementary material is available at Journal of the Ameri­ can Medical Informatics Association online. Funding This work was supported by National Institutes of Health grants: R00LM014097-02 and R01LM013995-01. Conflicts of interest The authors do not have conflicts of interest related to this study. Data availability The characteristics and outcomes for each included study were reported in the File S2. References 01. Raiaan MAK, Mukta MSH, Fatema K, et al. A review on large lan­ guage models: architectures, applications, taxonomies, open issues and challenges. IEEE Access. 2024;12:26839-26874. https://doi. org/10.1109/ACCESS.2024.3365742 02. Thirunavukarasu AJ, Ting DSJ, Elangovan K, et al. Large language models in medicine. Nat Med. 2023;29:1930-1940. https://doi. org/10.1038/s41591-023-02448-8 03. Ayers JW, Poliak A, Dredze M, et al. Comparing physician and arti­ ficial intelligence Chatbot responses to patient questions posted to a public social media forum. JAMA Intern Med. 2023;183:589-596. https://doi.org/10.1001/jamainternmed.2023.1838 04. Liu S, Wright AP, Patterson BL, et al. Using AI-generated sugges­ tions from ChatGPT to optimize clinical decision support. J Am Med Inform Assoc. 2023;30:1237-1245. https://doi.org/10.1093/ jamia/ocad072 05. Zaretsky J, Kim JM, Baskharoun S, et al. Generative artificial intel­ ligence to transform inpatient discharge summaries to patient- friendly language and format. JAMA Netw Open. 2024;7: e240357. https://doi.org/10.1001/jamanetworkopen.2024.0357 06. Gao Y, Xiong Y, Gao X, et al. Retrieval-augmented generation for large language models: a survey. 2023, preprint: not peer reviewed. https://arxiv.org/abs/2312.10997 07. Xu Z, Jain S, Kankanhalli M. Hallucination is inevitable: an innate limitation of large language models. January 22, 2024, preprint: not peer reviewed. https://arxiv.org/abs/2401.11817 08. Shuster K, Poff S, Chen M, et al. Retrieval augmentation reduces hallucination in conversation. Findings of the Association for Com­ putational Linguistics, Findings of ACL: EMNLP 2021. Associa­ tion for Computational Linguistics (ACL); 2021:3784-3803. 09. Ayala O, Bechard P. Reducing hallucination in structured outputs via Retrieval-Augmented Generation. Proceedings of the 2024 Conference of the North American Chapter of the Association for Computational Linguistics: Human Language Technologies. Association for Computational Linguistics; 2024;228-38. https:// doi.org/10.18653/v1/2024.naacl-industry.19 10. Gornik HL, Aronow HD, Goodney PP, et al. 2024 ACC/AHA/ AACVPR/APMA/ABC/SCAI/SVM/SVN/SVS/SIR/VESS guideline for the management of lower extremity peripheral artery disease: a report of the American College of Cardiology/American Heart Association Joint Committee on Clinical Practice Guidelines. Cir­ culation. 2024;149:e1313-e1410. https://doi.org/10.1161/CIR. 0000000000001251 11. Vasey B, Nagendran M, Campbell B, et al. Reporting guideline for the early stage clinical evaluation of decision support systems driven by artificial intelligence: DECIDE-AI. BMJ. 2022;377: e070904. https://doi.org/10.1136/bmj-2022-070904 12. Mongan J, Moy L, Kahn CE. Checklist for Artificial Intelligence in Medical Imaging (CLAIM): a guide for authors and reviewers. Radiol Artif Intell. 2020;2:e200029. https://doi.org/10.1148/ryai. 2020200029 13. Martindale APL, Ng B, Ngai V, et al. Concordance of randomised controlled trials for artificial intelligence interventions with the CONSORT-AI reporting guidelines. Nat Commun. 2024;15:6376-6311. https://doi.org/10.1038/s41467-024-45355-3 14. Tam TYC, Sivarajkumar S, Kapoor S, et al. A framework for human evaluation of large language models in healthcare derived from literature review. NPJ Digit Med. 2024;7:258. https://doi. org/10.1038/s41746-024-01258-7 15. Shamseer L, Moher D, Clarke M, et al. Preferred reporting items for systematic review and meta-analysis protocols (PRISMA-p) 2015: elaboration and explanation. BMJ. 2015;350:g7647. https://doi.org/10.1136/bmj.g7647 16. Higgins JPT, Thomas J, Chandler J, et al. (editors). Cochrane Hand­ book for Systematic Reviews of Interventions version 6.5 (updated August 2024). Cochrane, 2024. Available from www.training. cochrane.org/handbook. Date accessed December 11, 2024. Journal of the American Medical Informatics Association, 2025, Vol. 32, No. 4 613 Downloaded from https://academic.oup.com/jamia/article/32/4/605/7954485 by guest on 11 January 2026
{ "author": "", "creationDate": "D:20250322110116+05'30'", "creationdate": "2025-03-22T11:01:16+05:30", "creator": "Servigistics Arbortext Advanced Print Publisher 11.1.4667/W", "file_path": "/home/donbr/open-biosciences/biosciences-research/data/raw/ocaf008.pdf", "format": "PDF 1.5", "keywords": "large language model; retrieval augmented generation; systematic review; meta-analysis", "modDate": "D:20260111011327+00'00'", "moddate": "2026-01-11T01:13:27+00:00", "page": 8, "producer": "PDFlib+PDI 9.0.7p3 (C++/Win32); modified using iTextSharp.LGPLv2.Core 3.7.4.0", "source": "/home/donbr/open-biosciences/biosciences-research/data/raw/ocaf008.pdf", "subject": "Doi: 10.1093/jamia/ocaf008 Journal of the American Medical Informatics Association, 32, 4, 2025 Publication Date: 15/01/2025 Abstract Objective The objectives of this study are to synthesize findings from recent research of retrieval-augmented generation (RAG) and large language models (LLMs) in biomedicine and provide clinical development guidelines to improve effectiveness.Materials and Methods We conducted a systematic literature review and a meta-analysis. The report was created in adherence to the Preferred Reporting Items for Systematic Reviews and Meta-Analyses 2020 analysis. Searches were performed in 3 databases (PubMed, Embase, PsycINFO) using terms related to “retrieval augmented generation” and “large language model,” for articles published in 2023 and 2024. We selected studies that compared baseline LLM performance with RAG performance. We developed a random-effect meta-analysis model, using odds ratio as the effect size.Results Among 335 studies, 20 were included in this literature review. The pooled effect size was 1.35, with a 95% confidence interval of 1.19-1.53, indicating a statistically significant effect (P = .001). We reported clinical tasks, baseline LLMs, retrieval sources and strategies, as well as evaluation methods.Discussion Building on our literature review, we developed Guidelines for Unified Implementation and Development of Enhanced LLM Applications with RAG in Clinical Settings to inform clinical applications using RAG.Conclusion Overall, RAG implementation showed a 1.35 odds ratio increase in performance compared to baseline LLMs. Future research should focus on (1) system-level enhancement: the combination of RAG and agent, (2) knowledge-level enhancement: deep integration of knowledge into LLM, and (3) integration-level enhancement: integrating RAG systems within electronic health records.", "title": "Improving large language model applications in biomedicine with retrieval-augmented generation: a systematic review, meta-analysis, and clinical development guidelines", "total_pages": 11, "trapped": "" }
17. Lefebvre C, Glanville J, Briscoe S, et al. Chapter 4: searching for and selecting studies. Cochrane Handbook for Systematic Reviews of Interventions Version, Vol. 6. Cochrane, 2024. https://training. cochrane.org/handbook/current/chapter-04 18. Chapter 3 Effect Sizes j Doing Meta-Analysis in R. Accessed Octo­ ber 13, 2024. https://bookdown.org/MathiasHarrer/Doing_Meta_ Analysis_in_R/effects.html 19. Borenstein M, Hedges LV, Higgins JPT, et al. Introduction to Meta-Analysis. John Wiley & Sons; 2011. 20. Higgins JPT, Thompson SG. Quantifying heterogeneity in a meta- analysis. Stat Med. 2002;21:1539-1558. https://doi.org/10.1002/ sim.1186 21. Peters JL, Sutton AJ, Jones DR, et al. Contour-enhanced meta- analysis funnel plots help distinguish publication bias from other causes of asymmetry. J Clin Epidemiol. 2008;61:991-996. https:// doi.org/10.1016/j.jclinepi.2007.11.010 22. Egger M, Smith GD, Schneider M, et al. Bias in meta-analysis detected by a simple, graphical test measures of funnel plot asymmetry. BMJ. 1997;315:629-634. https://doi.org/10.1136/bmj.315.7109.629 23. Kreimeyer K, Canzoniero JV, Fatteh M, et al. Using retrieval- augmented generation to capture molecularly-driven treatment relationships for precision oncology. Stud Health Technol Inform. 2024;316:983-987. https://doi.org/10.3233/SHTI240575 24. Murugan M, Yuan B, Venner E, et al. Empowering personalized pharmacogenomics with generative AI solutions. J Am Med Inform Assoc. 2024;31:1356-1366. https://doi.org/10.1093/jamia/ocae039 25. Yazaki M, Maki S, Furuya T, et al. Emergency patient triage improvement through a retrieval-augmented generation enhanced large-scale language model. Prehosp Emerg Care. 2024;1-7. https://doi.org/10.1080/10903127.2024.2374400 26. Malik S, Kharel H, Dahiya DS, et al. Assessing ChatGPT4 with and without retrieval-augmented generation in anticoagulation management for gastrointestinal procedures. Ann Gastroenterol. 2024;37:514-526. https://doi.org/10.20524/aog.2024.0907 27. Kresevic S, Giuffre M, Ajcevic M, et al. Optimization of hepatolog­ ical clinical guidelines interpretation by large language models: a retrieval augmented generation-based framework. NPJ Digit Med. 2024;7:102-109. https://doi.org/10.1038/s41746-024-01091-y 28. Zakka C, Shad R, Chaurasia A, et al. Almanac–retrieval-aug­ mented language models for clinical medicine. NEJM AI. 2024;1 (2):10.1056/aioa2300068. https://doi.org/10.1056/aioa2300068 29. Long C, Subburam D, Lowe K, et al. ChatENT: augmented large language model for expert knowledge retrieval in otolaryngology– head and neck surgery. Otolaryngol Head Neck Surg. 2024;171:1042-1051. https://doi.org/10.1002/ohn.864 30. Wang D, Liang J, Ye J, et al. Enhancement of the performance of large language models in diabetes education through retrieval-aug­ mented generation: comparative study. J Med Internet Res. 2024;26:e58041. https://doi.org/10.2196/58041 31. Rau S, Rau A, Nattenm€uller J, et al. A retrieval-augmented chatbot based on GPT-4 provides appropriate differential diagnosis in gas­ trointestinal radiology: a proof of concept study. Eur Radiol Exp. 2024;8:60. https://doi.org/10.1186/s41747-024-00457-x 32. Morris JH, Soman K, Akbas RE, et al. The scalable precision medicine open knowledge engine (SPOKE): a massive knowl­ edge graph of biomedical information. Bioinformatics. 2023;39(2):btad080. https://doi.org/10.1093/BIOINFOR­ MATICS/BTAD080 33. Soman K, Rose PW, Morris JH, et al. Biomedical knowl­ edge graph-optimized prompt generation for large language models. Commun ACM. 2023;66:7-7. https://doi.org/10.1145/ 3606337 34. Jeong M, Sohn J, Sung M, et al. Improving medical reasoning through retrieval and self-reflection with retrieval-augmented large language models. Bioinformatics. 2024;40:i119-i129. https://doi. org/10.1093/bioinformatics/btae238 35. Alkhalaf M, Yu P, Yin M, et al. Applying generative AI with retrieval augmented generation to summarize and extract key clinical information from electronic health records. J Biomed Inform. 2024;156:104662. https://doi.org/10.1016/j.jbi.2024. 104662 36. Tarabanis C, Zahid S, Mamalis M, et al. Performance of publicly available large language models on internal medicine board-style questions. PLOS Digit Health. 2024;3:e0000604. https://doi.org/ 10.1371/journal.pdig.0000604 37. Glicksberg BS, Timsina P, Patel D, et al. Evaluating the accuracy of a state-of-the-art large language model for prediction of admis­ sions from the emergency room. J Am Med Inform Assoc. 2024;31:1921-1928. https://doi.org/10.1093/jamia/ocae103 38. Zelin C, Chung WK, Jeanne M, et al. Rare disease diagnosis using knowledge guided retrieval augmentation for ChatGPT. J Biomed Inform. 2024;157:104702. https://doi.org/10.1016/j.jbi.2024.104702 39. Chen X, Wang L, You MK, et al. Evaluating and enhancing large language models’ performance in domain-specific medicine: devel­ opment and usability study with DocOA. J Med Internet Res. 2024;26:e58158. https://doi.org/10.2196/58158 40. Du X, Novoa-Laurentiev J, Plasaek JM, et al. Enhancing early detection of cognitive decline in the elderly: a comparative study utilizing large language models in clinical notes. medRxiv. 2024;109:105401. https://doi.org/10.1101/2024.04.03.24305298 41. Zhang Y, Li Y, Cui L, et al. Siren’s song in the AI Ocean: a survey on hallucination in large language models. 2023, preprint: not peer reviewed. https://arxiv.org/abs/2309.01219 42. Li X, Ouyang JA. Systematic investigation of knowledge retrieval and selection for retrieval augmented generation. 2024, preprint: not peer reviewed. https://arxiv.org/abs/2410.13258 43. Xiong G, Jin Q, Lu Z, et al. Benchmarking retrieval-augmented generation for medicine. Findings of the Association for Computa­ tional Linguistics: ACL 2024, 6233-6251. Bangkok, Thailand: Association for Computational Linguistics. 44. Hewitt KJ, Wiest IC, Carrero ZI, et al. Large language models as a diagnostic support tool in neuropathology. J Pathol Clin Res. 2024;10:e70009. https://doi.org/10.1002/2056-4538.70009 45. Allahverdiyev R, Taha M, Akalin A, et al. ChunkRAG: novel LLM-chunk filtering method for RAG systems. October 25, 2024, preprint: not peer reviewed. https://arxiv.org/abs/2410.19572 46. Cai B, Zhang FL, Wang C. Research on chunking algorithms of data de-duplication. Advances in Intelligent Systems and Comput­ ing, Vol. 181. 2013:1019-1025. Springer, Berlin, Heidelberg. https://doi.org/10.1007/978-3-642-31698-2_144 47. Optimizing RAG with Advanced Chunking Techniques. Accessed December 15, 2024. https://antematter.io/blogs/optimizing-rag- advanced-chunking-techniques-study 48. Chen J, Xiao S, Zhang P, et al. M3-Embedding: multi-lingual, multi-functionality, multi-granularity text embeddings through self-knowledge distillation. Findings of the Association for Com­ putational Linguistics ACL 2024. 2024:2318-2335. Association for Computational Linguistics. https://doi.org/10.18653/V1/2024. FINDINGS-ACL.137 49. Sawarkar K, Mangal A, Solanki SR. Blended RAG: improving RAG (Retriever-Augmented Generation) accuracy with semantic search and hybrid query-based retrievers. 2024 IEEE 7th Interna­ tional Conference on Multimedia Information Processing and Retrieval (MIPR), 155-161. San Jose, CA, USA: IEEE. https://doi. org/10.1109/MIPR62202.2024.00031 50. Edge D, Trinh H, Cheng N, et al. From local to global: a graph RAG approach to query-focused summarization. 2024, preprint: not peer reviewed. https://arxiv.org/abs/2404.16130 51. Wu Z, Hasan A, Wu J, et al. KnowLab_AIMed at MEDIQA- CORR 2024: Chain-of-Though (CoT) prompting strategies for medical error detection and correction. Proceedings of the 6th Clinical Natural Language Processing Workshop. 2024:353-359. Association for Computational Linguistics. 10.18653/v1/2024. clinicalnlp-1.33 52. Kwon T, Tzu-Iunn Ong K, Kang D, et al. Large language models are clinical reasoners: reasoning-aware diagnosis framework with 614 Journal of the American Medical Informatics Association, 2025, Vol. 32, No. 4 Downloaded from https://academic.oup.com/jamia/article/32/4/605/7954485 by guest on 11 January 2026
{ "author": "", "creationDate": "D:20250322110116+05'30'", "creationdate": "2025-03-22T11:01:16+05:30", "creator": "Servigistics Arbortext Advanced Print Publisher 11.1.4667/W", "file_path": "/home/donbr/open-biosciences/biosciences-research/data/raw/ocaf008.pdf", "format": "PDF 1.5", "keywords": "large language model; retrieval augmented generation; systematic review; meta-analysis", "modDate": "D:20260111011327+00'00'", "moddate": "2026-01-11T01:13:27+00:00", "page": 9, "producer": "PDFlib+PDI 9.0.7p3 (C++/Win32); modified using iTextSharp.LGPLv2.Core 3.7.4.0", "source": "/home/donbr/open-biosciences/biosciences-research/data/raw/ocaf008.pdf", "subject": "Doi: 10.1093/jamia/ocaf008 Journal of the American Medical Informatics Association, 32, 4, 2025 Publication Date: 15/01/2025 Abstract Objective The objectives of this study are to synthesize findings from recent research of retrieval-augmented generation (RAG) and large language models (LLMs) in biomedicine and provide clinical development guidelines to improve effectiveness.Materials and Methods We conducted a systematic literature review and a meta-analysis. The report was created in adherence to the Preferred Reporting Items for Systematic Reviews and Meta-Analyses 2020 analysis. Searches were performed in 3 databases (PubMed, Embase, PsycINFO) using terms related to “retrieval augmented generation” and “large language model,” for articles published in 2023 and 2024. We selected studies that compared baseline LLM performance with RAG performance. We developed a random-effect meta-analysis model, using odds ratio as the effect size.Results Among 335 studies, 20 were included in this literature review. The pooled effect size was 1.35, with a 95% confidence interval of 1.19-1.53, indicating a statistically significant effect (P = .001). We reported clinical tasks, baseline LLMs, retrieval sources and strategies, as well as evaluation methods.Discussion Building on our literature review, we developed Guidelines for Unified Implementation and Development of Enhanced LLM Applications with RAG in Clinical Settings to inform clinical applications using RAG.Conclusion Overall, RAG implementation showed a 1.35 odds ratio increase in performance compared to baseline LLMs. Future research should focus on (1) system-level enhancement: the combination of RAG and agent, (2) knowledge-level enhancement: deep integration of knowledge into LLM, and (3) integration-level enhancement: integrating RAG systems within electronic health records.", "title": "Improving large language model applications in biomedicine with retrieval-augmented generation: a systematic review, meta-analysis, and clinical development guidelines", "total_pages": 11, "trapped": "" }
prompt-generated rationales. Proceedings of the AAAI Conference on Artificial Intelligence. Association for the Advancement of Arti­ ficial Intelligence; 2024:18417-18425. 53. Du X, Novoa-Laurentiev J, Plasek JM, et al. Enhancing early detection of cognitive decline in the elderly: a comparative study utilizing large language models in clinical notes. EBioMedicine. 2024;109:105401. https://doi.org/10.1016/j.ebiom.2024.105401 54. Embi PJ. Algorithmovigilance—advancing methods to analyze and monitor artificial intelligence–driven health care for effectiveness and equity. JAMA Netw Open. 2021;4:e214622. https://doi.org/ 10.1001/jamanetworkopen.2021.4622 55. Xi Z, Chen W, Guo X, et al. The rise and potential of large lan­ guage model based agents: a survey. September 14, 2023, preprint: not peer reviewed. 56. Wang L, Ma C, Feng X, et al. A survey on large language model based autonomous agents. Front Comput Sci. 2024;18:1-26. https://doi.org/10.1007/S11704-024-40231-1/METRICS 57. Li X, Wang S, Zeng S, et al. A survey on LLM-based multi-agent systems: workflow, infrastructure, and challenges. Vicinagearth. 2024;1:9. https://doi.org/10.1007/s44336-024-00009-2 58. Yang J, Shu L, Duan H, et al. RDguru: a conversational intelligent agent for rare diseases. IEEE J Biomed Health Inform. Published online September 19, 2024. https://doi.org/10.1109/JBHI.2024. 3464555 59. Ren R, Wang Y, Qu Y, et al. Investigating the factual knowledge boundary of large language models with retrieval augmentation. 2023, preprint: not peer reviewed. https://arxiv.org/abs/2307. 11019 60. Weed LL. Medical records that guide and teach. N Engl J Med. 1968;278:593-600. https://doi.org/10.1056/NEJM19680314278 1105/ASSET/9EE62BDC-88EB-469C-BCDC-DB379C2CAE47/ ASSETS/IMAGES/MEDIUM/NEJM196803142781105_F2.GIF 61. McCoy LG, Manrai AK, Rodman A. Large language mod­ els and the degradation of the medical record. N Engl J Med. 2024;391:1561-1564. https://doi.org/10.1056/ NEJMP2405999 62. Mandel JC, Kreda DA, Mandl KD, et al. SMART on FHIR: a standards-based, interoperable apps platform for electronic health records. J Am Med Inform Assoc. 2016;23:899-908. https://doi. org/10.1093/jamia/ocv189 © The Author(s) 2025. Published by Oxford University Press on behalf of the American Medical Informatics Association. This is an Open Access article distributed under the terms of the Creative Commons Attribution-NonCommercial License (https://creativecommons.org/licenses/by-nc/ 4.0/), which permits non-commercial re-use, distribution, and reproduction in any medium, provided the original work is properly cited. For commercial re-use, please contact reprints@oup.com for reprints and translation rights for reprints. All other permissions can be obtained through our RightsLink service via the Permissions link on the article page on our site—for further information please contact journals.permissions@oup.com. Journal of the American Medical Informatics Association, 2025, 32, 605–615 https://doi.org/10.1093/jamia/ocaf008 Research and Applications Journal of the American Medical Informatics Association, 2025, Vol. 32, No. 4 615 Downloaded from https://academic.oup.com/jamia/article/32/4/605/7954485 by guest on 11 January 2026
{ "author": "", "creationDate": "D:20250322110116+05'30'", "creationdate": "2025-03-22T11:01:16+05:30", "creator": "Servigistics Arbortext Advanced Print Publisher 11.1.4667/W", "file_path": "/home/donbr/open-biosciences/biosciences-research/data/raw/ocaf008.pdf", "format": "PDF 1.5", "keywords": "large language model; retrieval augmented generation; systematic review; meta-analysis", "modDate": "D:20260111011327+00'00'", "moddate": "2026-01-11T01:13:27+00:00", "page": 10, "producer": "PDFlib+PDI 9.0.7p3 (C++/Win32); modified using iTextSharp.LGPLv2.Core 3.7.4.0", "source": "/home/donbr/open-biosciences/biosciences-research/data/raw/ocaf008.pdf", "subject": "Doi: 10.1093/jamia/ocaf008 Journal of the American Medical Informatics Association, 32, 4, 2025 Publication Date: 15/01/2025 Abstract Objective The objectives of this study are to synthesize findings from recent research of retrieval-augmented generation (RAG) and large language models (LLMs) in biomedicine and provide clinical development guidelines to improve effectiveness.Materials and Methods We conducted a systematic literature review and a meta-analysis. The report was created in adherence to the Preferred Reporting Items for Systematic Reviews and Meta-Analyses 2020 analysis. Searches were performed in 3 databases (PubMed, Embase, PsycINFO) using terms related to “retrieval augmented generation” and “large language model,” for articles published in 2023 and 2024. We selected studies that compared baseline LLM performance with RAG performance. We developed a random-effect meta-analysis model, using odds ratio as the effect size.Results Among 335 studies, 20 were included in this literature review. The pooled effect size was 1.35, with a 95% confidence interval of 1.19-1.53, indicating a statistically significant effect (P = .001). We reported clinical tasks, baseline LLMs, retrieval sources and strategies, as well as evaluation methods.Discussion Building on our literature review, we developed Guidelines for Unified Implementation and Development of Enhanced LLM Applications with RAG in Clinical Settings to inform clinical applications using RAG.Conclusion Overall, RAG implementation showed a 1.35 odds ratio increase in performance compared to baseline LLMs. Future research should focus on (1) system-level enhancement: the combination of RAG and agent, (2) knowledge-level enhancement: deep integration of knowledge into LLM, and (3) integration-level enhancement: integrating RAG systems within electronic health records.", "title": "Improving large language model applications in biomedicine with retrieval-augmented generation: a systematic review, meta-analysis, and clinical development guidelines", "total_pages": 11, "trapped": "" }
Joy et. al 1 Federated Knowledge Retrieval Elevates Large Language Model Performance on Biomedical Benchmarks Janet Joy1,2, Andrew I. Su1,2 1Department of Integrative Structural and Computational Biology, Scripps Research, La Jolla, CA, USA. 2Correspondence: Janet Joy (jjoy@scripps.edu) and Andrew I. Su (asu@scripps.edu) Main Figures: 4 Supplemental Figures: 11 Supplemental Table: 1 Abstract Background Large language models (LLMs) have significantly advanced natural language processing in biomedical research, however, their reliance on implicit, statistical representations often results in factual inaccuracies or hallucinations, posing significant concerns in high-stakes biomedical contexts. Results To overcome these limitations, we developed BTE-RAG, a retrieval-augmented generation framework that integrates the reasoning capabilities of advanced language models with explicit mechanistic evidence sourced from BioThings Explorer, an API federation of more than sixty authoritative biomedical knowledge sources. We systematically evaluated BTE-RAG in comparison to traditional LLM-only methods across three benchmark datasets that we created from DrugMechDB. These datasets specifically targeted gene-centric mechanisms (798 questions), metabolite effects (201 questions), and drug–biological process relationships (842 questions). On the gene- centric task, BTE-RAG increased accuracy from 51% to 75.8% for GPT-4o mini and from 69.8% to 78.6% for GPT-4o. In metabolite-focused questions, the proportion of responses with cosine similarity scores of at least 0.90 rose by 82% for GPT-4o mini and 77% for GPT-4o. While overall accuracy was consistent in the drug–biological process benchmark, the retrieval method enhanced response concordance, producing a greater than 10% increase in high-agreement answers (from 129 to 144) using GPT-4o. Conclusion Federated knowledge retrieval provides transparent improvements in accuracy for large language models, establishing BTE-RAG as a valuable and practical tool for mechanistic exploration and translational biomedical research. . CC-BY 4.0 International license available under a (which was not certified by peer review) is the author/funder, who has granted bioRxiv a license to display the preprint in perpetuity. It is made The copyright holder for this preprint this version posted August 2, 2025. ; https://doi.org/10.1101/2025.08.01.668022 doi: bioRxiv preprint
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Joy et. al 2 1 Introduction Large language models (LLMs) have rapidly advanced the state of natural-language processing, reaching or surpassing expert performance across a wide range of biomedical tasks, including cell type annotation, protein-structure prediction and automated synthesis of clinical-trial results 1–6. However, the underlying generative methodology of these models, which sequentially predict tokens based on statistical patterns learned from massive text corpora, renders them susceptible to hallucinations, defined as outputs that are syntactically fluent yet factually incorrect 7,8. Such inaccuracies pose significant risks in biomedicine, where even minor errors can misdirect research efforts, delay critical therapeutic discoveries, or compromise patient safety 7,9–11. Indeed, recent assessments underscore that hallucination rates remain too high for safe and effective deployment in clinical and research-intensive environments 12,13. Efforts to mitigate these hallucinations through domain-specific pre-training and prompt engineering have yielded only incremental improvements, as these approaches continue to embed knowledge implicitly within opaque model parameters and fail to reliably surface evidence provenance 14–16. Retrieval-augmented generation (RAG) has emerged as a promising solution, explicitly grounding model-generated responses by dynamically incorporating external, verifiable evidence into prompts 17–19. Within biomedical question-answering contexts, RAG approaches consistently reduce hallucinations and elevate factual accuracy compared to parameter-only models. Nonetheless, the efficacy of RAG hinges critically on the precision, comprehensiveness, and currency of the retrieved contextual evidence 20–22. Knowledge graphs (KGs) are particularly compelling resources for RAG because they explicitly represent biological entities and their relationships, support multi-hop mechanistic reasoning, and maintain persistent identifiers that simplify provenance tracking 23–26. Yet most biomedical KGs are tuned to a narrow slice of biology (for example, protein–protein interactions) or require extensive curation to remain current, limiting their utility for cross-domain mechanistic reasoning. To address these challenges, BioThings Explorer (BTE) integrates and federates 61 authoritative biomedical APIs into a continuously updated meta-knowledge graph that encompasses genes, pathways, drugs, diseases, phenotypes, and more 27. The API-centric framework of BTE returns structured JSON triples annotated with semantic types and evidence citations from reputable biomedical databases such as Gene Ontology, DrugBank, and Pubmed central using Translator Reasoner API (TRAPI) specification 28–30. Here, we introduce BTE–RAG (BioThings Explorer–Retrieval-Augmented Generation), a novel framework that integrates the conversational fluency and reasoning capabilities of advanced LLMs with the explicit, multi-domain mechanistic knowledge captured by BTE. BTE–RAG dynamically executes targeted, query-focused graph traversals to retrieve concise, mechanistically pertinent evidence, formulates this evidence into declarative context statements, and augments model prompts accordingly. To rigorously assess the performance of BTE-RAG in biomedical question answering, we systematically created three specialized benchmark datasets from DrugMechDB, a curated knowledge base containing 5,666 expert-annotated mechanistic pathways with literature validation 31. These datasets consist of gene-centric (n = 798), metabolite- . CC-BY 4.0 International license available under a (which was not certified by peer review) is the author/funder, who has granted bioRxiv a license to display the preprint in perpetuity. It is made The copyright holder for this preprint this version posted August 2, 2025. ; https://doi.org/10.1101/2025.08.01.668022 doi: bioRxiv preprint
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Joy et. al 3 centric (n = 201), and drug-centric (n = 842) question–answer pairs, each explicitly reflecting the causal flow from drug through intermediate biological nodes to disease outcomes. Across all three DrugMechDB-derived benchmarks, BTE–RAG robustly improves factual grounding, accelerates convergence to correct responses over diverse biomedical entities relative to an LLM-only baseline. Collectively, these findings establish BTE–RAG as a powerful, practical tool for reducing hallucination risks and enhancing mechanistic clarity, significantly advancing the transparency, reliability, and utility of language model-driven biomedical discovery and clinical decision-making. 2 Materials & Methods 2.1 BTE–RAG Framework and Baseline Comparison The BTE–RAG framework systematically compares two distinct inference routes to evaluate the impact of structured, mechanistic context on large language model (LLM) outputs (Figure 1A). The first inference route, labeled "LLM-only," directly submits user- generated questions to the language model without external context augmentation. The second route, labeled "BTE–RAG," integrates structured mechanistic evidence retrieved from BioThings Explorer prior to submitting an enriched, evidence-supported prompt to the same language model. This dual-path design allows rigorous evaluation of how explicitly retrieved context influences both answer accuracy and the factual grounding of model-generated responses. The BTE–RAG architecture comprises three key phases: entity recognition, knowledge-graph-based retrieval via BTE, and generative inference utilizing context-augmented LLM prompting. Entity Recognition The retrieval phase begins with precise identification of biomedical entities mentioned within each input question. For the current benchmarks, entities such as drugs, diseases, metabolites, and biological processes were pre-annotated and standardized to established knowledge graph identifiers, enabling automated recognition at runtime. Additionally, the framework includes a zero-shot entity extraction module that leverages a specialized task-oriented prompting approach. This module is currently optimized for retrieving drugs and diseases from queries, with potential to extend extraction capabilities to include other biomedical entities as needed. Knowledge Graph Retrieval Identified biomedical entities are translated into structured queries interfacing directly with BTE. BTE integrates 61 authoritative biomedical databases under a unified knowledge graph schema, accessible via the programmatic API endpoint (/v1/query). Each query to BTE specifies an input entity (e.g., disease, drug, or biological process) along with desired output entity categories, following the TRAPI query format. In response, BTE returns structured JSON data that includes a detailed knowledge graph containing two key components: "nodes," which describe biomedical entities along with their semantic categories and standardized names; and "edges," which specify the . CC-BY 4.0 International license available under a (which was not certified by peer review) is the author/funder, who has granted bioRxiv a license to display the preprint in perpetuity. It is made The copyright holder for this preprint this version posted August 2, 2025. ; https://doi.org/10.1101/2025.08.01.668022 doi: bioRxiv preprint
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Joy et. al 4 explicit relationships (predicates) between pairs of entities, supplemented by provenance details indicating the primary knowledge sources. For each benchmark dataset, targeted queries were structured to retrieve mechanistically relevant context. Specifically, in the gene-centric benchmark, queries separately utilized disease and drug entities to retrieve directly linked gene and protein nodes. In the metabolite-centric benchmark, disease and chemical (drug) entities were queried independently to identify connected biochemical entities. For the drug– biological process benchmark, separate queries using disease entities and biological Figure 1: Retrieval‑Augmented Generation workflow and derivation of mechanistic evaluation benchmarks. (A) Schematic of the BTE-RAG pipeline, which augments large language model (LLM) responses with context retrieved from the BioThings Explorer (BTE) knowledge graph. In the LLM-only pathway, the model generates a response using only the input question. In contrast, BTE-RAG operates in two phases: a Retrieval Phase, where relevant entities are extracted from the question and queried against BTE to collect mechanistically relevant subject–predicate–object triples, and a Generative Phase, where this curated context is appended to the input question and passed to the LLM. The resulting outputs: LLM-only or BTE-RAG, can be directly compared to assess the impact of knowledge- augmented generation. (B) Construction of benchmark datasets from DrugMechDB, a curated biomedical knowledge graph of drug–disease mechanisms. Directed paths connecting a drug to a disease were mined and transformed into structured questions targeting different mechanistic facets: (i) gene nodes (Mechanistic Gene Benchmark), (ii) biochemical entities or metabolites (Metabolite Benchmark), and (iii) drug–biological process– disease paths (Drug Benchmark). Each benchmark provides paired questions and gold‑standard labels for rigorous, domain‑specific evaluation of retrieval‑augmented generation. . CC-BY 4.0 International license available under a (which was not certified by peer review) is the author/funder, who has granted bioRxiv a license to display the preprint in perpetuity. It is made The copyright holder for this preprint this version posted August 2, 2025. ; https://doi.org/10.1101/2025.08.01.668022 doi: bioRxiv preprint
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Joy et. al 5 process entities were conducted to retrieve associated chemical entities (drugs). Upon receiving the structured knowledge graph responses from BTE, both node and edge information were systematically processed. Nodes were extracted along with their semantic categories and descriptive names, while edges were parsed to identify subject-object pairs, predicates, and associated primary knowledge sources. Nodes and edges were subsequently merged to construct coherent statements that succinctly describe each mechanistic relationship (e.g., "drug X inhibits gene Y"). These concise, natural-language context statements collectively formed the mechanistic evidence provided to the language models during the generative inference phase, significantly enhancing the transparency, interpretability, and accuracy of the generated outputs. Supplementary Figure S1 provides a detailed schematic illustrating the complete BTE– RAG pipeline workflow, demonstrating a representative query and the subsequent processing and integration steps. Context Selection Two distinct evidence-inclusion strategies were systematically assessed for each question. The first strategy incorporates the entire set of sentences retrieved by BTE, leveraging the extensive 128,000-token context window of GPT-4o 32. The second strategy employs sentence-level cosine similarity filtering using ‘S-PubMedBert-MS- MARCO’ embeddings, retaining only sentences whose similarity scores with the query exceed a predefined percentile threshold 33. Running these two strategies concurrently enables a direct evaluation of the impact of comprehensive versus selectively pruned contextual evidence under identical experimental conditions. Generative Inference For the generative phase, selected context sentences and the original query were concatenated to form an enriched prompt submitted to both GPT-4o and GPT-4o-mini models. Models were configured deterministically (temperature set to 0) to produce reproducible outputs. Parallel runs of the LLM-only baseline used identical questions without the BTE-derived context. To streamline downstream analyses and ensure objective comparisons, language models were instructed explicitly to output structured JSON responses devoid of extraneous explanatory text. Detailed system prompts are shown in Supplementary Table S1. Evaluation Generated outputs were benchmarked against curated ground-truth annotations from the constructed mechanistic datasets. For entity-specific tasks (e.g., gene identification), correctness was evaluated via exact, case-insensitive string matching. For semantically nuanced responses (e.g., metabolites and drugs), BioBERT-based embeddings (‘BioBERT-mnli-snli-scinli-scitail-mednli-stsb’) quantified the semantic similarity between model-generated outputs and reference answers 34. Answers surpassing a predetermined similarity threshold were classified as accurate. Collectively, these standardized evaluation methodologies ensure scalable, objective, and reproducible assessment of the fidelity and biological coherence of model predictions, rigorously testing the utility and impact of knowledge graph-enhanced prompting in biomedical reasoning contexts. . CC-BY 4.0 International license available under a (which was not certified by peer review) is the author/funder, who has granted bioRxiv a license to display the preprint in perpetuity. It is made The copyright holder for this preprint this version posted August 2, 2025. ; https://doi.org/10.1101/2025.08.01.668022 doi: bioRxiv preprint
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Joy et. al 6 2.2 Datasets from DMDB 2.2 Construction of Mechanistic Question–Answer Benchmarks from DrugMechDB DrugMechDB is a rigorously curated biomedical knowledge graph designed to represent therapeutic mechanisms through explicit stepwise paths. These pathways originate from drug nodes, traverse biologically meaningful intermediate entities, and culminate at disease nodes, collectively delineating mechanisms underlying drug–disease interactions 31. The current version of DrugMechDB contains 5,666 curated mechanistic pathways, providing comprehensive coverage for 4,583 distinct drug–disease indications. Each node within DrugMechDB is systematically mapped to a standardized Biolink category and anchored to stable identifiers, while each relationship (edge) is annotated with a controlled predicate 35. This structured, granular, and provenance-rich resource enables robust benchmarking of computational models focused on mechanistic inference rather than simple associative or co-occurrence patterns. To comprehensively evaluate the BTE–RAG framework across multiple levels of biological resolution, DrugMechDB was systematically transformed into three complementary mechanistic question–answer (QA) benchmarks, each highlighting a distinct biological focus: genes, metabolites, and drugs (Figure 1B). Gene-Centric Benchmark: Mechanistic pathways were initially filtered to retain those containing exactly one internal node annotated as a Gene entity. Gene identifiers were resolved into standardized HGNC symbols using MyGene.info services; pathways containing deprecated or ambiguous identifiers were systematically excluded 36. Each remaining mechanistic pathway was converted into a structured question of the form: "Which gene plays the most significant mechanistic role in how Drug 'X' treats or impacts Disease 'Y'?" The corresponding HGNC gene symbol served as the definitive ground truth. Following deduplication across different indications, this dataset comprised 798 unique QA pairs. Metabolite-Centric Benchmark: To capture downstream biochemical effects, pathways exclusively containing taxonomic relationships (such as “subclass” predicates) were removed to ensure mechanistic specificity. Selected pathways included exactly one metabolite node, identified specifically by filtering node identifiers prefixed with "CHEBI:" to denote biochemical entities. Records containing multiple mechanistic pathways were excluded to maintain dataset simplicity and clarity. Each qualifying pathway was formulated into the structured question: "Which biochemical entity is affected by Drug 'X' via its mechanism of action in treating Disease 'Y'?" The metabolite node identified via CHEBI identifiers served as the ground truth answer, yielding a final dataset of 201 unique QA pairs. Drug-Centric Benchmark: A third benchmark dataset was developed to evaluate the ability of computational models to infer therapeutic agents when provided with a disease and a mediating biological process. Pathways were selected specifically if they included exactly one BiologicalProcess node, and drugs lacking resolvable identifiers from DrugBank or MESH databases were excluded to ensure accurate and standardized identification. Each qualifying path was structured into the question: "Which drug can be . CC-BY 4.0 International license available under a (which was not certified by peer review) is the author/funder, who has granted bioRxiv a license to display the preprint in perpetuity. It is made The copyright holder for this preprint this version posted August 2, 2025. ; https://doi.org/10.1101/2025.08.01.668022 doi: bioRxiv preprint
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Joy et. al 7 used in the treatment of Disease 'Y' by targeting Biological Process 'P'?" The corresponding drug node served as the ground truth. After thorough harmonization and stringent quality control measures, this benchmark comprised 842 unique QA pairs. The resulting benchmarks thus offer a robust, multiscale evaluation platform specifically designed to probe the mechanistic inference capabilities of knowledge-graph- augmented language models comprehensively and rigorously. 2.3 Use of Large Language Models: All natural-language processing steps were carried out with two OpenAI models, GPT-4o-mini (snapshot 2024-07-18) and GPT-4o (snapshot 2024-08-06) 32,37. Both models were invoked through the OpenAI API. The temperature parameter was fixed at 0.0 for every request, thereby forcing deterministic decoding and facilitating reproducible evaluation. Each model accepts up to 128,000 input tokens and can return a maximum of 16,384 completion tokens. Although GPT-4o-mini is substantially smaller in parameter count than GPT-4o, both models share the same context window size, permitting a controlled comparison of model capacity while holding prompt length constant 32,37. At the time the experiments were executed, GPT-4o-mini was priced at 0.15 USD per million input tokens and 0.60 USD per million output tokens. The corresponding prices for GPT-4o were 2.50 USD and 10.00 USD, respectively. Model versions were pinned by explicit snapshot identifiers to eliminate the possibility of version drift during the study period. Snapshot documentation is archived at https://platform.openai.com/docs/models/gpt-4o-mini and https://platform.openai.com/docs/models/gpt-4o. ChatGPT was used to assist with grammar correction and to improve conciseness in the manuscript. Prompt engineering Each request began with a concise system prompt defining the model’s role 38–40. Two distinct system prompts were prepared per dataset: one for the standalone LLM baseline, and one tailored for the retrieval-augmented BTE–RAG workflow. Queries were provided directly to the model without additional contextual examples, employing a zero-shot prompting approach. To facilitate efficient and accurate downstream processing, the model was instructed to produce responses strictly in a predefined JSON format, omitting supplementary explanatory text. 3 Results We developed BTE–RAG, a retrieval-augmented generation framework designed to enhance large language models (LLMs) by integrating mechanistic evidence from BioThings Explorer (BTE), a federated biomedical knowledge graph. BTE–RAG embeds structured, graph-derived context into prompts to improve mechanistic accuracy, ensure explicit provenance, and facilitate higher-order reasoning. We benchmarked the performance of BTE–RAG versus an LLM-only baseline across three distinct mechanistic reasoning tasks: gene identification, drug–metabolite interactions, and drug–biological-process relationships. 3.1 Mechanistic Gene Prediction . CC-BY 4.0 International license available under a (which was not certified by peer review) is the author/funder, who has granted bioRxiv a license to display the preprint in perpetuity. It is made The copyright holder for this preprint this version posted August 2, 2025. ; https://doi.org/10.1101/2025.08.01.668022 doi: bioRxiv preprint
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Joy et. al 8 We first assessed the effect of knowledge graph augmentation on gene-level mechanistic inference using 798 curated drug–disease pairs from DrugMechDB. Queries were structured as: "Which gene plays the most significant mechanistic role in how Drug 'X' treats or impacts Disease 'Y'?" Two models, GPT-4o and the smaller GPT- 4o-mini were evaluated in two experimental conditions: (i) LLM-only, providing no additional context, and (ii) BTE–RAG, incorporating evidence retrieved via BTE. Under the LLM-only condition, GPT-4o-mini correctly answered 407 queries (Supplementary Figure S2B), achieving an accuracy of 51%. Augmenting prompts with BTE-derived evidence markedly increased accuracy to 75.8% (Figure 2A), a substantial absolute improvement of 24.8 percentage points (Supplementary Figure S2C). The larger GPT-4o model demonstrated an accuracy of 69.8% in the baseline condition, which increased to 78.6% (627 correct answers, Supplementary Figure S3B) when supplemented with BTE context (Figure 2B), reflecting an absolute gain of 8.8 percentage points (Supplementary Figure S3C). Because knowledge‑graph queries can return superfluous triples, we evaluated a simple similarity‑based pruning strategy. Specifically, both the user queries and the context statements were embedded using the sentence embedding model 'S- PubMedBert-MS-MARCO' 33. Context statements were then ranked based on cosine Figure 2: Retrieval‑augmented generation with BTE-RAG markedly improves factual accuracy of gene-centric benchmark using GPT‑4o models. (A) For the compact gpt‑4o‑mini model, introducing the BTE-RAG retrieval layer raised overall accuracy from 51% (hatched bar, LLM‑only baseline) to 75.8 % (solid bar). (B) The same intervention applied to the larger gpt‑4o model increased accuracy from 69.8% to 78.6 %. Accuracy was calculated as the proportion of correct answers across the composite biomedical question‑answering benchmark described in Methods. . CC-BY 4.0 International license available under a (which was not certified by peer review) is the author/funder, who has granted bioRxiv a license to display the preprint in perpetuity. It is made The copyright holder for this preprint this version posted August 2, 2025. ; https://doi.org/10.1101/2025.08.01.668022 doi: bioRxiv preprint
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Joy et. al 9 similarity scores relative to the embedded query, and those statements falling within the lowest 10% similarity scores were removed to retain only the most relevant context lines. This lightweight filtering strategy preserved, and in some cases slightly enhanced performance across all evaluated accuracy metrics (Supplementary Figure S2A, S3A), suggesting that excluding the least relevant context statements can beneficially impact the accuracy of gene-level reasoning tasks. Cross-tabulation provided quantitative detail on the effect of retrieval augmentation, illustrating that BTE context flipped 245 previously incorrect answers to correct for GPT-4o-mini and 119 for GPT-4o (Supplementary Figure S2D, S3D). Together, these findings illustrate that structured mechanistic context provided through BTE significantly enhances gene-level reasoning performance, particularly amplifying the capabilities of smaller-scale models such as GPT-4o-mini. The accuracy improvements observed even in GPT-4o highlight that state-of-the-art models retain latent knowledge gaps effectively bridged by integrating curated biomedical graphs and selectively pruning irrelevant content. 3.2 Prediction of Drug–Metabolite Relationships To gauge whether retrieval augments the mechanistic fidelity of metabolite‑level reasoning, we posed 201 queries of the form “Which biochemical entity is affected by Drug X via its mechanism of action in treating Disease Y?” using the DrugMechDB‑ derived Drug → Metabolite → Disease paths. Because metabolite names are much less standardized than gene names, we scored the answer quality by computing a semantic concordance between each model answer and the gold standard metabolite. Semantic concordance was based on cosine similarity of text embeddings using the BioBERT- STSB text embedding model, a metric that rewards graded lexical and semantic overlap rather than exact string identity 34. Rank‑ordered similarity curves in Figure 3A immediately reveal the effect of augmentation: for both gpt‑4o‑mini (orange) and gpt‑4o (blue), the BTE‑RAG trace (solid line) departs from the prompt‑only baseline (dashed line) after ~130 ranked questions (cosine ≈ 0.70) and widens steadily, nearly doubling the number of answers that reach the high‑fidelity zone (cosine ≥ 0.90). Histograms for the prompt‑only condition (Figure 3B, gpt4o; Supplementary Figure S4, gpt-4o-mini) reveal a pronounced left‑skew: both gpt‑4o‑mini and gpt‑4o peak in the 0.15–0.30 similarity bins, with medians below 0.30. Only 15 % of answers fall in the high‑similarity regime (≥ 0.90), indicating that the LLMs frequently retrieve metabolites that are semantically distant from the curated ground truth. Appending BTE evidence shifts the distributions rightward across similarity bins (Figure 3C (gpt-4o), Supplementary Figure S5, S6). For GPT-4o-mini, applying a stringent context similarity threshold (>80th percentile) increased the number of high-fidelity answers (cosine similarity 0.90–1.00) from 28 to 51 (+82%). Similarly, GPT-4o exhibited an increase from 30 to 53 (+77%) under the same conditions. Simultaneously, counts in the mid‑similarity interval (0.40–0.70) contract (Supplementary Figure S5, S6), . CC-BY 4.0 International license available under a (which was not certified by peer review) is the author/funder, who has granted bioRxiv a license to display the preprint in perpetuity. It is made The copyright holder for this preprint this version posted August 2, 2025. ; https://doi.org/10.1101/2025.08.01.668022 doi: bioRxiv preprint
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Joy et. al 10 confirming that retrieval largely converts borderline predictions into highly concordant hits rather than merely redistributing low‑score failures. Because voluminous context can inflate token budgets, we assessed performance when progressively discarding lower‑ranked context lines (10th to 90th percentile cut‑offs). Rank‑ordered similarity traces (Supplementary Figure S7) show that the BTE‑RAG curves remain above or coincide with the prompt‑only baseline throughout the distribution even when 90 % of context is withheld. Histograms (Supplementary Figure S5, S6) reinforce this observation: the ≥ 0.90 similarity bin consistently retains Figure 3: Retrieval‑augmented context increases semantic concordance with ground‑truth metabolites. (A) Cosine‑similarity scores between each generated answer and the corresponding reference metabolite (sentence‑transformer embeddings; see Methods) are plotted for all 201 questions in the Metabolite Benchmark, ordered from lowest to highest similarity. Dashed traces represent the LLM‑only baseline, whereas solid traces include BioThings Explorer (BTE) retrieval‑augmented context. Orange curves denote gpt‑4o‑mini; blue curves denote gpt‑4o. For both model sizes, BTE‑RAG systematically shifts the similarity distribution upward, indicating improved semantic alignment with the curated biochemical ground truth. (B) Score distribution GPT-4o, LLM-only. Histogram of cosine-similarity scores for GPT-4o answers generated without external context. Bar heights and numeric labels denote the number of questions (n = 201) falling in each bin; the overlaid KDE line summarizes the distribution. (C) Score distribution GPT-4o + BTE-RAG. Same format as panel B but for GPT-4o answers generated with BTE-RAG's context. The right-shifted, more peaked distribution highlights the improvement in semantic alignment achieved by retrieval-augmented generation. . CC-BY 4.0 International license available under a (which was not certified by peer review) is the author/funder, who has granted bioRxiv a license to display the preprint in perpetuity. It is made The copyright holder for this preprint this version posted August 2, 2025. ; https://doi.org/10.1101/2025.08.01.668022 doi: bioRxiv preprint
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Joy et. al 11 ≥ 40 hits for both models across all pruning levels, demonstrating that a concise subset of top‑ranked evidence lines is sufficient to drive the bulk of the performance gains. 3.3 Drug–Biological Process Reasoning We next asked 842 DrugMechDB questions of the form “Which drug can be used in the treatment of Disease Y by targeting Biological Process P?”. Answer fidelity was again scored with BioBERT‑STSB cosine similarity 34. In rank‑ordered plots (Figure 4A), the prompt‑only (dashed) and BTE‑RAG (solid) curves for both gpt‑4o‑mini (orange) and Figure 4: Retrieval-augmented generation maintains overall parity yet excels in the high-fidelity regime of drug-centric mechanistic answers. (A) Cosine-similarity scores (sentence-transformer embeddings; see Methods) between each generated answer and the reference drug→biological-process pathway are plotted for all 842 questions in the Drug Benchmark, ordered from lowest to highest similarity. Dashed traces (LLM-only) and solid traces (BTE-RAG) follow nearly overlapping trajectories across most of the distribution, indicating broadly comparable performance between the two inference modes. However, above a cosine similarity threshold of ≈ 0.7, both gpt-4o-mini (orange) and gpt-4o (blue) curves generated with BTE context surge ahead of their prompt-only counterparts, revealing a marked advantage in producing highly concordant mechanistic explanations. (B) Score distribution GPT-4o, LLM-only. Histogram of cosine-similarity scores for GPT- 4o answers generated without external context. The hatched bar at 0.90–1.00 marks the high-fidelity zone, capturing 129 near-perfect matches produced by the baseline model. (C) Score distribution GPT-4o + BTE-RAG. Same format as panel B but for GPT-4o answers produced with BTE-RAG's context. The distribution is right-shifted, and the solid bar in the 0.90–1.00 high-fidelity zone now contains 144 answers, highlighting the enrichment of top-tier mechanistic concordance achieved through retrieval-augmented generation. . CC-BY 4.0 International license available under a (which was not certified by peer review) is the author/funder, who has granted bioRxiv a license to display the preprint in perpetuity. It is made The copyright holder for this preprint this version posted August 2, 2025. ; https://doi.org/10.1101/2025.08.01.668022 doi: bioRxiv preprint
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Joy et. al 12 gpt‑4o (blue) are nearly super‑imposable through the first ≈ 600 ranked queries (cosine < 0.70). Beyond this inflection point, the BTE‑augmented traces bend upward more steeply, yielding a clear margin in the high‑fidelity zone (cosine ≥ 0.80). Thus, retrieval does not alter overall parity but selectively boosts the most mechanistically demanding subset of questions. Prompt‑only histograms (Figure 4B; Supplementary Figure S8, gpt-4o-mini) peak in the 0.20–0.35 range, with ~15 % of answers falling in the ≥ 0.90 bin. Appending the full BTE context nudges the entire distribution rightward (Figure 4C; Supplementary Figure S9- top‑left panels). The ≥ 0.90 bin increases by ≈ 5–10 % for both model sizes. These shifts, though smaller than those seen for gene‑ and metabolite tasks, account for the late‑stage separation observed in Figure 4A. Unlike the previous tasks, performance here depends on retaining a broad evidentiary window. When the lowest‑ranked 10–20 % of context lines are removed, the uplift in the ≥ 0.90 bin attenuates, and the rank‑ordered curves progressively converge toward the baseline (Supplementary Figure S9, S10 & S11). Deeper cuts (> 40 %) essentially erase the retrieval advantage. This suggests that pathway‑level questions draw on a more diffuse set of graph triples than gene or metabolite queries, and aggressive trimming can discard critical relational clues. For drug → biological‑process reasoning, BTE‑RAG delivers targeted gains in the top decile of similarity scores, provided the complete knowledge‑graph context is supplied. These findings reinforce that optimal evidence granularity is task‑dependent: concise, high‑relevance snippets suffice for gene‑ and metabolite‑level inference, whereas pathway‑level queries benefit from a richer contextual fabric. By grounding LLM outputs within curated, biologically meaningful pathways, BTE–RAG consistently accelerates accurate inference, reduces residual errors, and demonstrates considerable promise for advancing automated biomedical hypothesis generation and therapeutic repurposing workflows. 4 Discussion The rapid advancement of large language models (LLMs) has profoundly reshaped biomedical natural language processing 41. Despite these advances, current LLMs predominantly operate as opaque systems with implicit knowledge representation, rendering their factual accuracy challenging to verify and limiting their applicability in high-stakes biomedical environments. Recent efforts, such as the knowledge-graph augmented retrieval approach 21, have successfully enhanced biomedical reasoning by integrating disease-specific embeddings from specialized knowledge graphs such as SPOKE 42. We developed BTE–RAG, a novel retrieval-augmented generation pipeline that strategically incorporates explicit mechanistic evidence from BTE 27. By leveraging the extensive and federated biomedical knowledge graph of BTE, our method substantially broadens the applicability of knowledge-graph augmented strategies to address diverse query types, including those involving genes, proteins, metabolites, biological processes, diseases and chemical substances. This capability allows BTE– RAG to support complex, multi-domain biomedical inquiries, significantly extending beyond disease-centric queries alone. Our comparative analysis, utilizing a direct "LLM- . CC-BY 4.0 International license available under a (which was not certified by peer review) is the author/funder, who has granted bioRxiv a license to display the preprint in perpetuity. It is made The copyright holder for this preprint this version posted August 2, 2025. ; https://doi.org/10.1101/2025.08.01.668022 doi: bioRxiv preprint
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Joy et. al 13 only" approach versus the BTE-augmented strategy (Figure 1A) across three rigorously constructed DrugMechDB benchmarks (Figure 1B), demonstrates that incorporating explicit, structured context significantly elevates answer accuracy, enhances transparency, and allows smaller, more computationally efficient models to perform competitively with leading-edge systems. The granularity, explicit mechanistic grounding, and high-quality source attribution inherent in these benchmarks uniquely position them for probing the causal inference capabilities of language models. Comparable mechanistically focused datasets remain scarce in the biomedical domain, as existing resources like PubMedQA or Natural Questions predominantly target document-level retrieval or summarization rather than deep mechanistic inference 43,44. Traditional LLMs accumulate domain-specific knowledge implicitly during pre-training by statistically modeling large collections of biomedical texts. Although this method yields linguistically coherent responses, it inherently exposes models to the risk of hallucinations, particularly in scenarios involving sparse biomedical facts or multi-step mechanistic reasoning. By contrast, retrieval-augmented generation explicitly anchors model predictions in verifiable external sources, constraining generation to well- substantiated evidence. BTE–RAG advances this paradigm by dynamically federating 61 authoritative biomedical APIs into a single cohesive meta-graph, thereby enabling real-time inclusion of newly curated knowledge in generated responses and ensuring reproducible benchmarking through cached retrievals. Four critical design principles underpin the efficacy of the BTE–RAG framework. First, the framework leverages an API-centric federation layer that integrates trusted biomedical data sources, including MyGene.info, Gene Ontology, CTDbase, Pubmed central, CHEBI, disease-ontology, DrugBank and more, through unified interface of BTE 29,36,45–47. Second, it employs semantic query templates aligned with the Translator Reasoner API (TRAPI) standard, selectively retrieving only the most relevant relationships for each question, thereby avoiding extraneous contextual noise. Third, retrieved knowledge graph triples are translated into succinct, directionally explicit declarative statements, seamlessly integrating structured knowledge with natural- language prompts. Fourth, BTE–RAG incorporates flexible context-selection strategies; full-context utilization and cosine similarity-based pruning for scenarios requiring concise, highly relevant context subsets. Across diverse mechanistic tasks, including gene-centric, metabolite-centric, and drug- centric benchmarks derived from DrugMechDB 31, BTE-augmented prompting consistently outperformed the LLM-only approach. Notably, the smaller GPT-4o-mini model achieved over sixty-percentage improvement in accuracy on the gene-centric task and eighty-two percent improvement on the metabolite task, when provided with structured BTE evidence. Even GPT-4o, the larger flagship model, demonstrated substantial accuracy gains, underscoring that high-quality, explicit mechanistic context can effectively mitigate the need for extremely large model sizes, suggesting a cost- efficient pathway toward domain-specific accuracy. While BTE offers comprehensive coverage across numerous biomedical domains, certain areas such as single-cell data, epigenomic profiles, and microbiome interactions remain sparsely represented. Furthermore, variations in curation quality across . CC-BY 4.0 International license available under a (which was not certified by peer review) is the author/funder, who has granted bioRxiv a license to display the preprint in perpetuity. It is made The copyright holder for this preprint this version posted August 2, 2025. ; https://doi.org/10.1101/2025.08.01.668022 doi: bioRxiv preprint
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Joy et. al 14 federated APIs could inadvertently propagate erroneous edges into model-generated contexts. Although our evaluation leveraged the meticulously curated, high-confidence knowledge graph of DrugMechDB, real-world applications may require strategies for managing lower-confidence or conflicting evidence. Our study employed deterministic prompting to maintain comparability; exploring guided, chain-of-thought prompting strategies could further enhance complex reasoning capabilities but may simultaneously reintroduce hallucinatory risks. Future developments of BTE–RAG may involve integration into autonomous agent systems capable of iterative querying, generation, self-critiquing, and re-querying, thus facilitating automated self-verification workflows. Expanding the underlying knowledge graph to incorporate resources such as LINCS transcriptomic signatures, tissue-specific interaction networks, and multi-omics datasets would further enrich the mechanistic coverage and broaden applicability 48. Expanding benchmarking efforts beyond DrugMechDB to encompass open-world biomedical queries could rigorously evaluate and strengthen the capacity BTE–RAG for reliable, contextually grounded inference. Furthermore, adopting frameworks like the Model Context Protocol could harmonize comparisons across diverse generative models, facilitate rigorous auditing, and support real-time decision-making in clinical and regulatory contexts. In conclusion, BTE–RAG demonstrates the substantial value derived from strategically integrating explicit mechanistic evidence into biomedical language modeling workflows. By significantly improving answer accuracy, interpretability, and computational efficiency, this approach provides a scalable, transparent, and robust foundation for future biomedical AI systems, effectively balancing accuracy, affordability, and trustworthiness. Supplementary File: Supplementary Figure S1: Detailed pipeline for BTE-RAG Supplementary Figure S2: Performance of BTE‑RAG versus an LLM‑only baseline on the gene‑centric benchmark using gpt‑4o‑mini. Supplementary Figure S3: Performance of BTE‑RAG versus an LLM‑only baseline on the gene‑centric benchmark using gpt‑4o. Supplementary Figure S4: Cosine-similarity profile for the metabolite-centric benchmark using GPT-4o-mini in LLM-only mode. Supplementary Figure S5: Distribution of answer similarities for the metabolite-centric benchmark using GPT-4o-mini in BTE-RAG mode. Supplementary Figure S6: Distribution of answer similarities for the metabolite-centric benchmark using GPT-4o in BTE-RAG mode. Supplementary Figure S7: Rank-ordered cosine similarities between model predictions and ground-truth answers on the metabolite-centric benchmark, across context filtering thresholds. . CC-BY 4.0 International license available under a (which was not certified by peer review) is the author/funder, who has granted bioRxiv a license to display the preprint in perpetuity. It is made The copyright holder for this preprint this version posted August 2, 2025. ; https://doi.org/10.1101/2025.08.01.668022 doi: bioRxiv preprint
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Joy et. al 15 Supplementary Figure S8: Cosine-similarity profile for the drug-centric benchmark using GPT-4o-mini in LLM-only mode. Supplementary Figure S9: Distribution of answer similarities for the drug-centric benchmark using GPT-4o-mini in BTE-RAG mode. Supplementary Figure S10: Distribution of answer similarities for the drug-centric benchmark using GPT-4o in BTE-RAG mode. Supplementary Figure S11: Rank-ordered cosine similarities between model predictions and ground-truth answers on the drug-centric benchmark, across context filtering thresholds. Supplementary Table S1: System prompts used for each task and model Availability of Data and Materials The source code, datasets, and analysis workflows described in this manuscript are publicly available in the GitHub repository: Project Name: BTE-RAG. Repository URL: https://github.com/janjoy/BTE-RAG Author contributions J.J. and A.S. conceived the project and proposed the benchmark curation. J.J. implemented the code, created the benchmarks and wrote the manuscript. A.S. supervised the study. All authors read and approved the final manuscript. Acknowledgements We thank Jackson Callaghan, Mikhael Astorga, and Karthik Soman for insightful discussions, and Everaldo Rodolpho for technical support with high-performance computing resources and server infrastructure. Competing Interests The authors declare no competing interests. Funding Support for this work was provided by the National Institute on Aging (award R01AG066750) and by the National Center for Advancing Translational Sciences through the Biomedical Data Translator program (awards 1OT2TR003427 and 1OT2TR005710). Any opinions expressed in this document do not necessarily reflect . CC-BY 4.0 International license available under a (which was not certified by peer review) is the author/funder, who has granted bioRxiv a license to display the preprint in perpetuity. It is made The copyright holder for this preprint this version posted August 2, 2025. ; https://doi.org/10.1101/2025.08.01.668022 doi: bioRxiv preprint
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Joy et. al 1 Supplementary Figures Figure S1: Detailed pipeline for BTE-RAG Supplementary Figure S1 depicts the end-to-end workflow through which the BTE-RAG retrieval module converts a biomedical question into the evidence snippets ultimately supplied to the language-model reasoner. Beginning with an example query, “Which gene plays the most significant mechanistic role in how the drug hydrochlorothiazide treats or impacts the disease hypertension?”, the system first performs named-entity recognition, normalizing the detected concepts to controlled identifiers (Drug: MESH:D006852; Disease: MESH:D006973). Each entity is then submitted to BioThings Explorer (BTE) as part of a query graph that requests mechanistically relevant genes and proteins; independent queries are executed for the drug and for the disease. BTE returns knowledge-graph sub-graphs whose nodes and edges represent subject-predicate-object triples grounded in the biomedical literature. These triples are linearized into plain-text sentences, yielding two preliminary corpora (one for the disease, one for the drug) that list, for example, genes such as INS, REN, SLC12A3 and PTGS2 with their associated predicates. Finally, the complete set of sentences or a percentile-filtered subset is ranked by cosine similarity to the original question, and the highest-scoring lines are selected as the “retrieved context” passed forward for answer generation. . CC-BY 4.0 International license available under a (which was not certified by peer review) is the author/funder, who has granted bioRxiv a license to display the preprint in perpetuity. It is made The copyright holder for this preprint this version posted August 2, 2025. ; https://doi.org/10.1101/2025.08.01.668022 doi: bioRxiv preprint
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