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Integrating de novo and inherited variants in 42,607 autism cases identifies mutations in new moderate-risk genes
Journal article   Open access

Integrating de novo and inherited variants in 42,607 autism cases identifies mutations in new moderate-risk genes

Xueya Zhou, Pamela Feliciano, Chang Shu, Tianyun Wang, Irina Astrovskaya, Jacob B Hall, Joseph U Obiajulu, Jessica R Wright, Shwetha C Murali, Simon Xuming Xu, …
Nature genetics, Vol.54(9), pp.1305-1319
08/18/2022
DOI: 10.1038/s41588-022-01148-2
PMCID: PMC9470534
PMID: 35982159
url
https://doi.org/10.1038/s41588-022-01148-2View
Published (Version of record) Open Access

Abstract

To capture the full spectrum of genetic risk for autism, we performed a two-stage analysis of rare de novo and inherited coding variants in 42,607 autism cases, including 35,130 new cases recruited online by SPARK. We identified 60 genes with exome-wide significance (P < 2.5 × 10−6), including five new risk genes (NAV3, ITSN1, MARK2, SCAF1 and HNRNPUL2). The association of NAV3 with autism risk is primarily driven by rare inherited loss-of-function (LoF) variants, with an estimated relative risk of 4, consistent with moderate effect. Autistic individuals with LoF variants in the four moderate-risk genes (NAV3, ITSN1, SCAF1 and HNRNPUL2; n = 95) have less cognitive impairment than 129 autistic individuals with LoF variants in highly penetrant genes (CHD8, SCN2A, ADNP, FOXP1 and SHANK3) (59% vs 88%, P = 1.9 × 10−6). Power calculations suggest that much larger numbers of autism cases are needed to identify additional moderate-risk genes.

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