Prioritizing non-coding regions based on human genomic constraint and sequence context with deep learning

Dimitrios Vitsios, Ryan S. Dhindsa, Lawrence Middleton, Ayal B. Gussow, Slavé Petrovski

Nature Communications 2021 article cited by 88

importance 7/10 partially solved Genomics and Rare Diseases genomics stage: research

Identifying which non-coding regions of the human genome are functional and disease-relevant is a key challenge in genomics.

Prioritizing disease-relevant non-coding variants can improve diagnosis of Mendelian and other diseases.

JARVIS, a deep learning model integrating intolerance-to-variation (gwRVIS from 62,784 individuals), functional annotations and genomic sequence, prioritizes non-coding regions.

Outperformed other human lineage-specific scores; performed comparably to or better than conservation-based scores for classifying pathogenic single-nucleotide and structural variants.

Provides a score that outperforms existing methods for prioritizing disease-relevant non-coding variants.

Venue Nature Communications
Published 2021
Work type article
Authors Dimitrios Vitsios, Ryan S. Dhindsa, Lawrence Middleton, Ayal B. Gussow, Slavé Petrovski
Citations 88 (OpenAlex, at import time)
References 50
Model or method JARVIS (deep learning model)
Organization not stated in source
Primary source https://doi.org/10.1038/s41467-021-21790-4
Open access gold
Keywords deep learning, non-coding regions, genomics, JARVIS, variant prioritization, gwRVIS
7 / 10

significant — thousands to millions affected

Non-coding functional annotation is a key human genomics challenge affecting disease-variant prioritization; JARVIS outperforms existing scores using 62,784 genomes, but no disease-population scale is stated.

Basis: inferred. Judged from the source text, which gives no explicit scale. Stated scale: not stated in source. Method and limits.

# Paper Year Citations Importance
1 AlphaFold Protein Structure Database: massively expanding the structural coverage of protein-sequence space with high-accuracy models
Mihály Váradi, Stephen Anyango, Mandar Deshpande, Sreenath Sasidharan...
2021 7,782
9
2 Evolutionary-scale prediction of atomic-level protein structure with a language model
Zeming Lin, Halil Akin, Roshan Rao, Brian Hie, Zhongkai Zhu, Wenting L...
2023 5,455
9
3 Accurate prediction of protein structures and interactions using a three-track neural network
Minkyung Baek, Frank DiMaio, Ivan V. Anishchenko, Justas Dauparas, Ser...
2021 5,468
9
4 De novo design of protein structure and function with RFdiffusion
Joseph L. Watson, David Juergens, Nathaniel R. Bennett, Brian L. Tripp...
2023 2,294
9
5 Structure Prediction and Computational Protein Design for Efficient Biocatalysts and Bioactive Proteins
Rebecca Buller, Jiřı́ Damborský, Donald Hilvert, Uwe T. Bornscheuer
2024 40
9