Analysis of protein-coding genetic variation in 60,706 humans

Анализ генетической вариабельности белок-кодирующих участков у 60 706 человек
Mark I. McCarthy, Stacey Gabriel, Gad Getz, Diego Ardissino, James Zou, Michael Boehnke, Steven A. McCarroll, Shaun Purcell, Benjamin M. Neale, Mark J. Daly, D.N. Cooper, Eric Banks, David Altshuler, Monkol Lek, Konrad J. Karczewski, Eric Vallabh Minikel, Kaitlin E. Samocha, Timothy R. Fennell, Anne O’Donnell‐Luria, James S. Ware, Andrew Hill, Beryl B. Cummings, Taru Tukiainen, Daniel P. Birnbaum, Jack A. Kosmicki, Laramie E. Duncan, Karol Estrada, Fengmei Zhao, Emma Pierce‐Hoffman, Joanne Berghout, Nicole Deflaux, Mark A. DePristo, Ron Do, Jason Flannick, Menachem Fromer, Laura D. Gauthier, Jackie Goldstein, Namrata Gupta, Daniel P. Howrigan, Adam Kieżun, Mitja Kurki, Ami Levy Moonshine, Pradeep Natarajan, Lorena Orozco, Gina M. Peloso, Ryan Poplin, Manuel A. Rivas, Valentín Ruano-Rubio, Samuel A. Rose, Douglas M. Ruderfer, Khalid Shakir, Peter D. Stenson, Christine Stevens, Brett Thomas, Grace Tiao, Maria T. Tusie-Luna, Ben Weisburd, Hong‐Hee Won, Dongmei Yu, John Danesh, Stacey Donnelly, Roberto Elosúa, José C. Florez, Stephen J. Glatt, Christina M. Hultman, Sekar Kathiresan, Markku Laakso, Dermot McGovern, Ruth McPherson, Aarno Palotie, Danish Saleheen, Jeremiah M. Scharf, Pamela Sklar, Patrick F. Sullivan, Jaakko Tuomilehto, Ming T. Tsuang, Hugh Watkins, James G. Wilson, Daniel G. MacArthur, Stacey Gabriel
2016-08-01

Exome Aggregation Consortiumexome sequencingpathogenicity metricsprotein-coding genetic variationprotein-truncating variants
Large-scale reference data sets of human genetic variation are critical for the medical and functional interpretation of DNA sequence changes. Here we describe the aggregation and analysis of high-quality exome (protein-coding region) DNA sequence data for 60,706 individuals of diverse ancestries generated as part of the Exome Aggregation Consortium (ExAC). This catalogue of human genetic diversity contains an average of one variant every eight bases of the exome, and provides direct evidence for the presence of widespread mutational recurrence. We have used this catalogue to calculate objective metrics of pathogenicity for sequence variants, and to identify genes subject to strong selection against various classes of mutation; identifying 3,230 genes with near-complete depletion of predicted protein-truncating variants, with 72% of these genes having no currently established human disease phenotype. Finally, we demonstrate that these data can be used for the efficient filtering of candidate disease-causing variants, and for the discovery of human ‘knockout’ variants in protein-coding genes. Exome sequencing data from 60,706 people of diverse geographic ancestry is presented, providing insight into genetic variation across populations, and illuminating the relationship between DNA variants and human disease. As part of the Exome Aggregation Consortium (ExAC) project, Daniel MacArthur and colleagues report on the generation and analysis of high-quality exome sequencing data from 60,706 individuals of diverse ancestry. This provides the most comprehensive catalogue of human protein-coding genetic variation to date, yielding unprecedented resolution for the analysis of very rare variants across multiple human populations. The catalogue is freely accessible and provides a critical reference panel for the clinical interpretation of genetic variants and the discovery of disease-related genes.
1
Among 3,230 genes showing near-complete depletion of predicted protein-truncating variants, 72% lack an established human disease phenotype.
2
ExAC enables objective pathogenicity metrics for sequence variants and reveals strong selection against multiple mutation classes in human genes.
3
The ExAC resource aggregates high-quality exome data from 60,706 individuals of diverse ancestries, creating a comprehensive catalogue of human protein-coding variation.
4
The catalogue identifies approximately one genetic variant every eight exome bases and provides direct evidence that mutational recurrence is widespread.
5
The resource supports efficient filtering of candidate disease-causing variants and discovery of human protein-coding gene knockout variants.

Aggregated high-quality human exome (protein-coding) sequence data from 60,706 individuals (ExAC)

The distribution, recurrence, pathogenicity, selection, and disease relevance of protein-coding genetic variants across human populations

Publication Details
Publication Date
2016-08-01
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Authors
Mark I. McCarthy
Stacey Gabriel
Gad Getz
Diego Ardissino
James Zou
Michael Boehnke
Steven A. McCarroll
Shaun Purcell
Benjamin M. Neale
Mark J. Daly
D.N. Cooper
Eric Banks
David Altshuler
Monkol Lek
Konrad J. Karczewski
Eric Vallabh Minikel
Kaitlin E. Samocha
Timothy R. Fennell
Anne O’Donnell‐Luria
James S. Ware
Andrew Hill
Beryl B. Cummings
Taru Tukiainen
Daniel P. Birnbaum
Jack A. Kosmicki
Laramie E. Duncan
Karol Estrada
Fengmei Zhao
Emma Pierce‐Hoffman
Joanne Berghout
Nicole Deflaux
Mark A. DePristo
Ron Do
Jason Flannick
Menachem Fromer
Laura D. Gauthier
Jackie Goldstein
Namrata Gupta
Daniel P. Howrigan
Adam Kieżun
Mitja Kurki
Ami Levy Moonshine
Pradeep Natarajan
Lorena Orozco
Gina M. Peloso
Ryan Poplin
Manuel A. Rivas
Valentín Ruano-Rubio
Samuel A. Rose
Douglas M. Ruderfer
Khalid Shakir
Peter D. Stenson
Christine Stevens
Brett Thomas
Grace Tiao
Maria T. Tusie-Luna
Ben Weisburd
Hong‐Hee Won
Dongmei Yu
John Danesh
Stacey Donnelly
Roberto Elosúa
José C. Florez
Stephen J. Glatt
Christina M. Hultman
Sekar Kathiresan
Markku Laakso
Dermot McGovern
Ruth McPherson
Aarno Palotie
Danish Saleheen
Jeremiah M. Scharf
Pamela Sklar
Patrick F. Sullivan
Jaakko Tuomilehto
Ming T. Tsuang
Hugh Watkins
James G. Wilson
Daniel G. MacArthur
Stacey Gabriel
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