Deep structured learning for variant prioritization in Mendelian diseases

MC Danzi, MF Dohrn, S Fazal, D Beijer… - Nature …, 2023 - nature.com
Effective computer-aided or automated variant evaluations for monogenic diseases will
expedite clinical diagnostic and research efforts of known and novel disease-causing genes …

DeepPVP: phenotype-based prioritization of causative variants using deep learning

I Boudellioua, M Kulmanov, PN Schofield… - BMC …, 2019 - Springer
Background Prioritization of variants in personal genomic data is a major challenge.
Recently, computational methods that rely on comparing phenotype similarity have shown to …

Disease variant prediction with deep generative models of evolutionary data

J Frazer, P Notin, M Dias, A Gomez, JK Min, K Brock… - Nature, 2021 - nature.com
Quantifying the pathogenicity of protein variants in human disease-related genes would
have a marked effect on clinical decisions, yet the overwhelming majority (over 98%) of …

A phenotype centric benchmark of variant prioritisation tools

D Anderson, T Lassmann - NPJ genomic medicine, 2018 - nature.com
Next generation sequencing is a standard tool used in clinical diagnostics. In Mendelian
diseases the challenge is to discover the single etiological variant among thousands of …

DANN: a deep learning approach for annotating the pathogenicity of genetic variants

D Quang, Y Chen, X Xie - Bioinformatics, 2014 - academic.oup.com
Annotating genetic variants, especially non-coding variants, for the purpose of identifying
pathogenic variants remains a challenge. Combined annotation-dependent depletion …

Updated benchmarking of variant effect predictors using deep mutational scanning

BJ Livesey, JA Marsh - Molecular systems biology, 2023 - embopress.org
The assessment of variant effect predictor (VEP) performance is fraught with biases
introduced by benchmarking against clinical observations. In this study, building on our …

DOMINO: using machine learning to predict genes associated with dominant disorders

M Quinodoz, B Royer-Bertrand, K Cisarova… - The American Journal of …, 2017 - cell.com
In contrast to recessive conditions with biallelic inheritance, identification of dominant
(monoallelic) mutations for Mendelian disorders is more difficult, because of the abundance …

[HTML][HTML] Improved pathogenicity prediction for rare human missense variants

Y Wu, H Liu, R Li, S Sun, J Weile, FP Roth - The American Journal of …, 2021 - cell.com
The success of personalized genomic medicine depends on our ability to assess the
pathogenicity of rare human variants, including the important class of missense variation …

MVP predicts the pathogenicity of missense variants by deep learning

H Qi, H Zhang, Y Zhao, C Chen, JJ Long… - Nature …, 2021 - nature.com
Accurate pathogenicity prediction of missense variants is critically important in genetic
studies and clinical diagnosis. Previously published prediction methods have facilitated the …

An improved phenotype-driven tool for rare mendelian variant prioritization: benchmarking exomiser on real patient whole-exome data

V Cipriani, N Pontikos, G Arno, PI Sergouniotis… - Genes, 2020 - mdpi.com
Next-generation sequencing has revolutionized rare disease diagnostics, but many patients
remain without a molecular diagnosis, particularly because many candidate variants usually …