Exome Sequencing of Two Siblings with Sporadic Autism Spectrum Disorder and Severe Speech Sound Disorder Suggests Pleiotropic and Complex Effects.
Adolescent
Adult
Autism Spectrum Disorder
/ etiology
Child
DNA Copy Number Variations
/ genetics
Exome
/ genetics
Family
Female
GTP-Binding Proteins
/ genetics
Genetic Pleiotropy
/ genetics
Genetic Predisposition to Disease
/ genetics
Humans
Male
Multifactorial Inheritance
/ genetics
Nerve Tissue Proteins
/ genetics
Siblings
Speech Sound Disorder
/ etiology
Tumor Suppressor Proteins
/ genetics
WW Domain-Containing Oxidoreductase
/ genetics
Exome Sequencing
/ methods
Autism spectrum disorder
Childhood apraxia of speech
Comorbidity
Complex trait
Heterogeneity
Language impairment
Multiple hits
Journal
Behavior genetics
ISSN: 1573-3297
Titre abrégé: Behav Genet
Pays: United States
ID NLM: 0251711
Informations de publication
Date de publication:
07 2019
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