Understanding genetic variants in context

Over the last three decades, human genetics has gone from dissecting high-penetrance Mendelian diseases to discovering the vast and complex genetic etiology of common human diseases. In tackling this complexity, scientists have discovered the importance of numerous genetic processes – most notably f...

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Main Authors: Nasa Sinnott-Armstrong, Stanley Fields, Frederick Roth, Lea M Starita, Cole Trapnell, Judit Villen, Douglas M Fowler, Christine Queitsch
Format: Article
Language:English
Published: eLife Sciences Publications Ltd 2024-12-01
Series:eLife
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Online Access:https://elifesciences.org/articles/88231
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author Nasa Sinnott-Armstrong
Stanley Fields
Frederick Roth
Lea M Starita
Cole Trapnell
Judit Villen
Douglas M Fowler
Christine Queitsch
author_facet Nasa Sinnott-Armstrong
Stanley Fields
Frederick Roth
Lea M Starita
Cole Trapnell
Judit Villen
Douglas M Fowler
Christine Queitsch
author_sort Nasa Sinnott-Armstrong
collection DOAJ
description Over the last three decades, human genetics has gone from dissecting high-penetrance Mendelian diseases to discovering the vast and complex genetic etiology of common human diseases. In tackling this complexity, scientists have discovered the importance of numerous genetic processes – most notably functional regulatory elements – in the development and progression of these diseases. Simultaneously, scientists have increasingly used multiplex assays of variant effect to systematically phenotype the cellular consequences of millions of genetic variants. In this article, we argue that the context of genetic variants – at all scales, from other genetic variants and gene regulation to cell biology to organismal environment – are critical components of how we can employ genomics to interpret these variants, and ultimately treat these diseases. We describe approaches to extend existing experimental assays and computational approaches to examine and quantify the importance of this context, including through causal analytic approaches. Having a unified understanding of the molecular, physiological, and environmental processes governing the interpretation of genetic variants is sorely needed for the field, and this perspective argues for feasible approaches by which the combined interpretation of cellular, animal, and epidemiological data can yield that knowledge.
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spelling doaj-art-69da816b45074542be41e4914ffd34732025-08-20T02:52:42ZengeLife Sciences Publications LtdeLife2050-084X2024-12-011310.7554/eLife.88231Understanding genetic variants in contextNasa Sinnott-Armstrong0https://orcid.org/0000-0003-4490-0601Stanley Fields1https://orcid.org/0000-0001-5504-5925Frederick Roth2Lea M Starita3Cole Trapnell4https://orcid.org/0000-0002-8105-4347Judit Villen5Douglas M Fowler6https://orcid.org/0000-0001-7614-1713Christine Queitsch7https://orcid.org/0000-0002-0905-4705Herbold Computational Biology Program, Fred Hutchinson Cancer Center, Seattle, United States; Department of Genome Sciences, University of Washington, Seattle, United States; Brotman Baty Institute for Precision Medicine, Seattle, United StatesDepartment of Genome Sciences, University of Washington, Seattle, United States; Department of Medicine, University of Washington, Seattle, United StatesDonnelly Centre and Departments of Molecular Genetics and Computer Science, University of Toronto, Toronto, Canada; Lunenfeld-Tanenbaum Research Institute, Mt. Sinai Hospital, Toronto, Canada; Department of Computational and Systems Biology, University of Pittsburgh School of Medicine, Pittsburgh, United StatesDepartment of Genome Sciences, University of Washington, Seattle, United States; Brotman Baty Institute for Precision Medicine, Seattle, United StatesDepartment of Genome Sciences, University of Washington, Seattle, United States; Brotman Baty Institute for Precision Medicine, Seattle, United StatesDepartment of Genome Sciences, University of Washington, Seattle, United States; Brotman Baty Institute for Precision Medicine, Seattle, United StatesDepartment of Genome Sciences, University of Washington, Seattle, United States; Brotman Baty Institute for Precision Medicine, Seattle, United States; Department of Bioengineering, University of Washington, Seattle, United StatesDepartment of Genome Sciences, University of Washington, Seattle, United States; Brotman Baty Institute for Precision Medicine, Seattle, United StatesOver the last three decades, human genetics has gone from dissecting high-penetrance Mendelian diseases to discovering the vast and complex genetic etiology of common human diseases. In tackling this complexity, scientists have discovered the importance of numerous genetic processes – most notably functional regulatory elements – in the development and progression of these diseases. Simultaneously, scientists have increasingly used multiplex assays of variant effect to systematically phenotype the cellular consequences of millions of genetic variants. In this article, we argue that the context of genetic variants – at all scales, from other genetic variants and gene regulation to cell biology to organismal environment – are critical components of how we can employ genomics to interpret these variants, and ultimately treat these diseases. We describe approaches to extend existing experimental assays and computational approaches to examine and quantify the importance of this context, including through causal analytic approaches. Having a unified understanding of the molecular, physiological, and environmental processes governing the interpretation of genetic variants is sorely needed for the field, and this perspective argues for feasible approaches by which the combined interpretation of cellular, animal, and epidemiological data can yield that knowledge.https://elifesciences.org/articles/88231gene–environment interactionsmultiplexed assays of variant effectepistasis
spellingShingle Nasa Sinnott-Armstrong
Stanley Fields
Frederick Roth
Lea M Starita
Cole Trapnell
Judit Villen
Douglas M Fowler
Christine Queitsch
Understanding genetic variants in context
eLife
gene–environment interactions
multiplexed assays of variant effect
epistasis
title Understanding genetic variants in context
title_full Understanding genetic variants in context
title_fullStr Understanding genetic variants in context
title_full_unstemmed Understanding genetic variants in context
title_short Understanding genetic variants in context
title_sort understanding genetic variants in context
topic gene–environment interactions
multiplexed assays of variant effect
epistasis
url https://elifesciences.org/articles/88231
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AT juditvillen understandinggeneticvariantsincontext
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