Behavior genetics · 2019
Exome Sequencing of Two Siblings with Sporadic Autism Spectrum Disorder and Severe Speech Sound Disorder Suggests Pleiotropic and Complex Effects.
Клинический случайСтатьяПоддержка исследования: вне правительства СШАНазвание и аннотация: Перевод в очереди
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Peter Beate, Dinu Valentin, Liu Li, Huentelman Matthew, Naymik Marcus, Lancaster Hope, Vose Caitlin, Schrauwen Isabelle
Аннотация
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.
Предметные рубрики MeSH
AdolescentAdultAutism Spectrum DisorderChildDNA Copy Number VariationsExomeFamilyFemaleGTP-Binding ProteinsGenetic PleiotropyGenetic Predisposition to DiseaseHumansMaleMultifactorial InheritanceNerve Tissue ProteinsSiblingsSpeech Sound DisorderTumor Suppressor ProteinsWW Domain-Containing OxidoreductaseExome Sequencing