Evidence map›Paper›PMID 42177353›Full record

ArticleScientific reports2026

Semantic embedding of variant effect annotations enables rapid and accurate pathogenicity prediction with VUS.Life.

Jiawei Wu, Marissa Stutzman, Michael Muriello, Joy Lincoln, Donald G Basel, Xiaowu Gai

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Article in Scientific reports, 2026. The graph could read no effect estimate from its abstract, so it casts no vote on the map. Not yet cited in PubMed.

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5 · Who and what money

Authors and funding

6 authors.

Jiawei WuDepartment of Pediatrics, Medical College of Wisconsin, Milwaukee, WI, 53226, USA.
Marissa StutzmanDepartment of Pediatrics, Medical College of Wisconsin, Milwaukee, WI, 53226, USA.
Michael MurielloDepartment of Pediatrics, Medical College of Wisconsin, Milwaukee, WI, 53226, USA.
Joy LincolnDepartment of Pediatrics, Medical College of Wisconsin, Milwaukee, WI, 53226, USA.
Donald G BaselDepartment of Pediatrics, Medical College of Wisconsin, Milwaukee, WI, 53226, USA.
Xiaowu GaiDepartment of Pediatrics, Medical College of Wisconsin, Milwaukee, WI, 53226, USA. xgai@mcw.edu.

Funding

No grant is acknowledged in the PubMed record.

6 · The paper itself

Abstract

Interpreting the pathogenicity of genetic variants remains a critical bottleneck in genomic medicine. Millions of variants of uncertain significance (VUS) hinder the clinical application of genetic findings. Traditional computational approaches often rely on hand-engineered features and fail to capture the complexity of multidimensional genomic annotations fully. We developed VUS.Life, a multi-modal framework that synergizes semantic text embeddings of biological and clinical annotations with protein language modeling. We transformed variant annotations from Variant Effect Predictor (VEP) into natural language descriptions which are then converted into vector embeddings via established Large Language Models (LLMs), namely all- mpnet-base-v2, MedEmbed-large-v0.1, and text-embedding-004. Pathogenicity of a variant of interest is predicted by its proximity in the vector embedding space with variants of known pathogenicity. We further extended VUS.Life by employing residue-level delta embeddings from the ESMC-600 M model to capture both clinical context and biophysical constraints. We evaluated the framework on > 10,000 variants across eight ACMG Tier 1 disease genes (BRCA1, BRCA2, FBN1, ATM, PALB2, MYH7, USH2A, and PAH), achieving Leave-One-Out Cross-Validation (LOO-CV) MCC of 0.895-0.989 and F1 ≥ 0.94 across all genes evaluated. Additionally, our unsupervised FBN1 structural analysis using ESMC-600M revealed that delta embeddings disentangled distinct pathogenic mechanisms, topologically separating disulfide bond disruptions from calcium- binding defects. These structural clusters correlated strongly with Zero-Shot Log-Likelihood Ratio (LLR) scores, validating evolutionary fitness as a proxy for pathogenicity. An ablation study removing all pre-computed pathogenicity scores demonstrated that MPNet embeddings retain full discriminative power, confirming that the classifier captures biological signal independent of existing scoring tools. This semantic embedding framework, VUS.Life, accurately captures pathogenicity-relevant features from complex variant annotations, enabling robust automated classification across eight ACMG Tier 1 disease genes and three embedding models. The approach generalizes beyond well-curated genes and supports scalable, interpretable, and representation-based classification of VUS. It holds significant promise for alleviating the variant interpretation bottleneck in clinical genomics.

Indexed as

Computational BiologyGenetic VariationMolecular Sequence AnnotationGenomicsHumansLarge Language ModelsSemanticsATMBRCA1BRCA2ESMC-600 MFBN1Large language modelMYH7PAHPALB2Pathogenicity predictionProtein structureUSH2AVector embedding

Identifiers

PMID42177353
PMCPMC13421479

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Read under generation 80e0d062 · epoch 390. Bibliography from PubMed, PubMed Central and OpenAlex; grants from NIH RePORTER; trial links from ClinicalTrials.gov; estimates, votes and beliefs from the OpenQuestion graph.