Gene-centered representation of coding and regulatory variation enables outcome prediction

Integrating coding and regulatory variation into unified, interpretable representations remains a challenge in functional genomics. Current approaches either focus on common variants or analyze individual variants in isolation, missing the cumulative, cell-type-specific impact of both coding and noncoding variants on each gene. We present Volaria, a computational framework that integrates coding and regulatory genetic variation into unified, gene-centered representations for disease outcome prediction from whole-genome sequencing. Volaria leverages deep learning models to capture variant effects on cell-type-specific gene expression and integrates them with AI-predicted exonic variant pathogenicity to produce representations that capture the cumulative effect of genome-wide rare and common variation. Applied to whole genomes of individuals with rare glomerular diseases, Volaria predicts individual outcomes directly from germline sequence, demonstrating that structured, cell-type-aware representations capture predictive signals beyond population-based polygenic risk scores and unstructured representations. Importantly, the framework identifies context-specific biological mechanisms, providing interpretability that can be aligned with clinical measurements. By encoding genome-wide variation into compact and biologically grounded representations, Volaria provides a scalable foundation for genome interpretation and individualized outcome modeling from germline sequence, complementing phenotypic and clinical information in the future integrative frameworks.

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Publication Details

Journal
bioRxiv (Cold Spring Harbor Laboratory)
Published
2026-01-29
DOI
https://doi.org/10.64898/2026.01.27.701808
Primary Topic
Genomics and Rare Diseases
Type
article
Field-Weighted Citation Impact
0.00

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article

Gene-centered representation of coding and regulatory variation enables outcome prediction

Chandra L. Theesfeld, Ksenia Sokolova, VN Kristensen, Laura Mariani et al.
bioRxiv (Cold Spring Harbor Laboratory)
Genomics and Rare Diseases
article

Gene-centered representation of coding and regulatory variation enables outcome prediction

Chandra L. Theesfeld, Ksenia Sokolova, VN Kristensen, Laura Mariani, Olga G Troyanskaya, Matthias Kretzler, Christopher Y Park
article en

Abstract

Integrating coding and regulatory variation into unified, interpretable representations remains a challenge in functional genomics. Current approaches either focus on common variants or analyze individual variants in isolation, missing the cumulative, cell-type-specific impact of both coding and noncoding variants on each gene. We present Volaria, a computational framework that integrates coding and regulatory genetic variation into unified, gene-centered representations for disease outcome prediction from whole-genome sequencing. Volaria leverages deep learning models to capture variant effects on cell-type-specific gene expression and integrates them with AI-predicted exonic variant pathogenicity to produce representations that capture the cumulative effect of genome-wide rare and common variation. Applied to whole genomes of individuals with rare glomerular diseases, Volaria predicts individual outcomes directly from germline sequence, demonstrating that structured, cell-type-aware representations capture predictive signals beyond population-based polygenic risk scores and unstructured representations. Importantly, the framework identifies context-specific biological mechanisms, providing interpretability that can be aligned with clinical measurements. By encoding genome-wide variation into compact and biologically grounded representations, Volaria provides a scalable foundation for genome interpretation and individualized outcome modeling from germline sequence, complementing phenotypic and clinical information in the future integrative frameworks.

bioRxiv (Cold Spring Harbor Laboratory)
Oslo University Hospital (NO), University of Oslo (NO), Princeton University (US), Simons Foundation (US), Michigan Medicine (US)
Princeton University, National Institutes of Health, National Institute of Diabetes and Digestive and Kidney Diseases
Openalex Percentile: Top 68%
Genomics and Rare Diseases
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