Guidelines for releasing a variant effect predictor

Abstract Computational methods for assessing the likely impacts of mutations, known as variant effect predictors (VEPs), are widely used in the assessment and interpretation of human genetic variation, as well as in other applications like protein engineering. Many different VEPs have been released,...

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Main Authors: Benjamin J. Livesey, Mihaly Badonyi, Mafalda Dias, Jonathan Frazer, Sushant Kumar, Kresten Lindorff-Larsen, David M. McCandlish, Rose Orenbuch, Courtney A. Shearer, Lara Muffley, Julia Foreman, Andrew M. Glazer, Ben Lehner, Debora S. Marks, Frederick P. Roth, Alan F. Rubin, Lea M. Starita, Joseph A. Marsh
Format: Article
Language:English
Published: BMC 2025-04-01
Series:Genome Biology
Online Access:https://doi.org/10.1186/s13059-025-03572-z
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author Benjamin J. Livesey
Mihaly Badonyi
Mafalda Dias
Jonathan Frazer
Sushant Kumar
Kresten Lindorff-Larsen
David M. McCandlish
Rose Orenbuch
Courtney A. Shearer
Lara Muffley
Julia Foreman
Andrew M. Glazer
Ben Lehner
Debora S. Marks
Frederick P. Roth
Alan F. Rubin
Lea M. Starita
Joseph A. Marsh
author_facet Benjamin J. Livesey
Mihaly Badonyi
Mafalda Dias
Jonathan Frazer
Sushant Kumar
Kresten Lindorff-Larsen
David M. McCandlish
Rose Orenbuch
Courtney A. Shearer
Lara Muffley
Julia Foreman
Andrew M. Glazer
Ben Lehner
Debora S. Marks
Frederick P. Roth
Alan F. Rubin
Lea M. Starita
Joseph A. Marsh
author_sort Benjamin J. Livesey
collection DOAJ
description Abstract Computational methods for assessing the likely impacts of mutations, known as variant effect predictors (VEPs), are widely used in the assessment and interpretation of human genetic variation, as well as in other applications like protein engineering. Many different VEPs have been released, and there is tremendous variability in their underlying algorithms, outputs, and the ways in which the methodologies and predictions are shared. This leads to considerable difficulties for users trying to navigate the selection and application of VEPs. Here, to address these issues, we provide guidelines and recommendations for the release of novel VEPs.
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publisher BMC
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spelling doaj-art-5a5f3378d6334c3cbfe4228efab46ae52025-08-20T02:17:47ZengBMCGenome Biology1474-760X2025-04-0126111310.1186/s13059-025-03572-zGuidelines for releasing a variant effect predictorBenjamin J. Livesey0Mihaly Badonyi1Mafalda Dias2Jonathan Frazer3Sushant Kumar4Kresten Lindorff-Larsen5David M. McCandlish6Rose Orenbuch7Courtney A. Shearer8Lara Muffley9Julia Foreman10Andrew M. Glazer11Ben Lehner12Debora S. Marks13Frederick P. Roth14Alan F. Rubin15Lea M. Starita16Joseph A. Marsh17MRC Human Genetics Unit, Institute of Genetics and Cancer, University of EdinburghMRC Human Genetics Unit, Institute of Genetics and Cancer, University of EdinburghCentre for Genomic Regulation (CRG), The Barcelona Institute of Science and TechnologyCentre for Genomic Regulation (CRG), The Barcelona Institute of Science and TechnologyDepartment of Medical Biophysics, University of TorontoDepartment of Biology, Linderstrøm-Lang Centre for Protein Science, University of CopenhagenSimons Center for Quantitative Biology, Cold Spring Harbor Laboratory, Cold Spring HarborDepartment of Systems Biology, Harvard Medical SchoolDepartment of Systems Biology, Harvard Medical SchoolDepartment of Genome Sciences, University of Washingtonand the, Brotman Baty Institute for Precision Medicine European Molecular Biology Laboratory, European Bioinformatics Institute, Wellcome Genome CampusVanderbilt University Medical CenterWellcome Sanger InstituteDepartment of Systems Biology, Harvard Medical SchoolDepartment of Computational and Systems Biology, University of Pittsburgh School of MedicineBioinformatics Division, Walterand , Eliza Hall Institute of Medical ResearchDepartment of Genome Sciences, University of Washingtonand the, Brotman Baty Institute for Precision Medicine MRC Human Genetics Unit, Institute of Genetics and Cancer, University of EdinburghAbstract Computational methods for assessing the likely impacts of mutations, known as variant effect predictors (VEPs), are widely used in the assessment and interpretation of human genetic variation, as well as in other applications like protein engineering. Many different VEPs have been released, and there is tremendous variability in their underlying algorithms, outputs, and the ways in which the methodologies and predictions are shared. This leads to considerable difficulties for users trying to navigate the selection and application of VEPs. Here, to address these issues, we provide guidelines and recommendations for the release of novel VEPs.https://doi.org/10.1186/s13059-025-03572-z
spellingShingle Benjamin J. Livesey
Mihaly Badonyi
Mafalda Dias
Jonathan Frazer
Sushant Kumar
Kresten Lindorff-Larsen
David M. McCandlish
Rose Orenbuch
Courtney A. Shearer
Lara Muffley
Julia Foreman
Andrew M. Glazer
Ben Lehner
Debora S. Marks
Frederick P. Roth
Alan F. Rubin
Lea M. Starita
Joseph A. Marsh
Guidelines for releasing a variant effect predictor
Genome Biology
title Guidelines for releasing a variant effect predictor
title_full Guidelines for releasing a variant effect predictor
title_fullStr Guidelines for releasing a variant effect predictor
title_full_unstemmed Guidelines for releasing a variant effect predictor
title_short Guidelines for releasing a variant effect predictor
title_sort guidelines for releasing a variant effect predictor
url https://doi.org/10.1186/s13059-025-03572-z
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