학술논문

Genomic architecture of autism from comprehensive whole-genome sequence annotation.
Document Type
article
Author
Trost, BrettThiruvahindrapuram, BhoomaChan, AdaEngchuan, WorrawatHigginbotham, EdwardHowe, JenniferLoureiro, LiviaReuter, MiriamRoshandel, DelnazWhitney, JoeZarrei, MehdiBookman, MatthewSomerville, CherithShaath, RulanAbdi, MonaAliyev, ElbayPatel, RohanNalpathamkalam, ThomasPellecchia, GiovannaHamdan, OmarKaur, GaganjotWang, ZhuozhiMacDonald, JeffreyWei, JohnSung, WilsonLamoureux, SylviaHoang, NySelvanayagam, ThanujaDeflaux, NicoleGeng, MelissaGhaffari, SiavashBates, JohnYoung, EdwinDing, QiliangShum, CaroleDAbate, LiaBradley, ClarrisaRutherford, AnnabelAguda, VernieApresto, BeverlyChen, NanDesai, SachinDu, XiaoyanFong, MatthewPullenayegum, SanjeevSamler, KozueWang, TingHo, KarenPaton, TaraPereira, SergioHerbrick, Jo-AnneWintle, RichardFuerth, JonathanNoppornpitak, JutiWard, HeatherMagee, PatrickAl Baz, AymanKajendirarajah, UsanthanKapadia, SharvariVlasblom, JimValluri, MonicaGreen, JosephSeifer, VickiQuirbach, MorganRennie, OliviaKelley, ElizabethMasjedi, NinaLord, CatherineSzego, MichaelZawati, ManLang, MichaelStrug, LisaMarshall, ChristianCostain, GregoryCalli, KristinaIaboni, AlanaYusuf, AfiqahAmbrozewicz, PatriciaGallagher, LouiseAmaral, DavidBrian, JessicaElsabbagh, MayadaGeorgiades, SteliosMessinger, DanielSebat, JonathanSjaarda, CalvinSmith, IsabelSzatmari, PeterZwaigenbaum, LonnieKushki, AzadehFrazier, ThomasVorstman, JacobFakhro, KhalidFernandez, BridgetLewis, MWeksberg, RosannaFiume, MarcYuen, RyanAnagnostou, EvdokiaSondheimer, Neal
Source
Cell. 185(23)
Subject
autism spectrum disorder
copy-number variation
neurodevelopmental disorders
phenotype measures
polygenic risk scores
rare variants
structural variation
whole-genome sequencing
Humans
Autism Spectrum Disorder
Autistic Disorder
Genetic Predisposition to Disease
DNA Copy Number Variations
Genomics
Language
Abstract
Fully understanding autism spectrum disorder (ASD) genetics requires whole-genome sequencing (WGS). We present the latest release of the Autism Speaks MSSNG resource, which includes WGS data from 5,100 individuals with ASD and 6,212 non-ASD parents and siblings (total n = 11,312). Examining a wide variety of genetic variants in MSSNG and the Simons Simplex Collection (SSC; n = 9,205), we identified ASD-associated rare variants in 718/5,100 individuals with ASD from MSSNG (14.1%) and 350/2,419 from SSC (14.5%). Considering genomic architecture, 52% were nuclear sequence-level variants, 46% were nuclear structural variants (including copy-number variants, inversions, large insertions, uniparental isodisomies, and tandem repeat expansions), and 2% were mitochondrial variants. Our study provides a guidebook for exploring genotype-phenotype correlations in families who carry ASD-associated rare variants and serves as an entry point to the expanded studies required to dissect the etiology in the ∼85% of the ASD population that remain idiopathic.