Exome Sequencing of Two Siblings with Sporadic Autism Spectrum Disorder and Severe Speech Sound Disorder Suggests Pleiotropic and Complex Effects.


Journal

Behavior genetics
ISSN: 1573-3297
Titre abrégé: Behav Genet
Pays: United States
ID NLM: 0251711

Informations de publication

Date de publication:
07 2019
Historique:
received: 25 11 2017
accepted: 18 03 2019
pubmed: 6 4 2019
medline: 14 2 2020
entrez: 6 4 2019
Statut: ppublish

Résumé

Recent studies of autism spectrum disorder (ASD) and childhood apraxia of speech (CAS) have resulted in conflicting conclusions regarding the comorbidity of these disorders on phenotypic grounds. In a nuclear family with two dually affected and one unaffected offspring, whole-exome sequences were evaluated for single nucleotide and indel variants and CNVs. The affected siblings but not the unaffected sibling share a rare deleterious compound heterozygous mutation in WWOX, implicated both in ASD and motor control. In addition, one of the affected children carries a rare deleterious de novo mutation in the ASD candidate gene RIMS1. The two affected children but not their unaffected sibling inherited deleterious variants with relevance for ASD and/or CAS. WWOX, RIMS1, and several of the genes harboring the inherited variants are expressed in the brain during prenatal and early postnatal development. Results suggest compound heterozygosity as a cause of ASD and CAS, pleiotropic gene effects, and potentially additional, complex genetic effects.

Identifiants

pubmed: 30949922
doi: 10.1007/s10519-019-09957-8
pii: 10.1007/s10519-019-09957-8
doi:

Substances chimiques

Nerve Tissue Proteins 0
RIMS1 protein, human 0
Tumor Suppressor Proteins 0
WW Domain-Containing Oxidoreductase EC 1.1.1.-
WWOX protein, human EC 1.1.1.-
GTP-Binding Proteins EC 3.6.1.-

Types de publication

Case Reports Journal Article Research Support, Non-U.S. Gov't

Langues

eng

Sous-ensembles de citation

IM

Pagination

399-414

Auteurs

Beate Peter (B)

Department of Speech and Hearing Science, Arizona State University, 975 S. Myrtle Ave, Tempe, AZ, 85287-0102, USA. Beate.Peter@asu.edu.
Department of Communication Sciences and Disorders, Saint Louis University, Saint Louis, MO, USA. Beate.Peter@asu.edu.

Valentin Dinu (V)

Department of Biomedical Informatics, Arizona State University, Tempe, AZ, USA.

Li Liu (L)

Department of Biomedical Informatics, Arizona State University, Tempe, AZ, USA.

Matthew Huentelman (M)

Neurogenomics Division, Translational Genomics Research Institute, Phoenix, AZ, USA.

Marcus Naymik (M)

Neurogenomics Division, Translational Genomics Research Institute, Phoenix, AZ, USA.

Hope Lancaster (H)

Department of Speech and Hearing Science, Arizona State University, 975 S. Myrtle Ave, Tempe, AZ, 85287-0102, USA.

Caitlin Vose (C)

Department of Speech and Hearing Science, Arizona State University, 975 S. Myrtle Ave, Tempe, AZ, 85287-0102, USA.

Isabelle Schrauwen (I)

Neurogenomics Division, Translational Genomics Research Institute, Phoenix, AZ, USA.
Center for Statistical Genetics, Department of Molecular and Human Genetics, Baylor College of Medicine, Houston, TX, USA.

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Classifications MeSH