Manning Christina, Hurd Peter L, Read Silven, Crespi Bernard
Department of Biological Sciences, Simon Fraser University, Burnaby, BC, Canada.
Department of Psychology and Centre for Neuroscience, University of Alberta, Edmonton, Canada.
Autism Res Treat. 2021 Jun 3;2021:6634584. doi: 10.1155/2021/6634584. eCollection 2021.
Mutations affecting the synaptic-scaffold gene SHANK3 represent the most common genetic causes of autism with intellectual disability, accounting for about 1-2% of cases. Rare variants of this gene have also been associated with schizophrenia, and its deletion results in the autistic condition known as Phelan-McDermid syndrome. Despite the importance of SHANK3 as a paradigmatic gene mediating neurodevelopmental disorders, its psychological effects in nonclinical populations have yet to be studied. We genotyped the nonsynonymous, functional SHANK3 SNP rs9616915 in a large population of typical individuals scored for autism spectrum traits (the Autism Quotient, AQ) and schizotypy spectrum traits (the Schizotypal Personality Questionnaire, SPQ-BR). Males, but not females, showed significant genotypic effects for the SPQ-BR subscale associated with speech and language: Odd Speech. These findings, in conjunction with animal model studies showing vocalization and auditory effects of SHANK3 mutations, and studies indicating severe language alterations and speech-associated white matter tract abnormalities in Phelan-McDermid syndrome, suggest that SHANK3 differentially affects the development and expression of human language and speech. Imaging genetic and speech-language studies of typical individuals carrying different genotypes of rs9616915 should provide novel insights into the neurological and psychological bases of speech and language alterations among individuals with SHANK3 mutations and Phelan-McDermid syndrome.
影响突触支架基因SHANK3的突变是导致伴有智力障碍的自闭症最常见的遗传原因,约占病例的1%-2%。该基因的罕见变异也与精神分裂症有关,其缺失会导致一种名为费伦-麦克德米德综合征的自闭症。尽管SHANK3作为介导神经发育障碍的典型基因很重要,但其在非临床人群中的心理影响尚未得到研究。我们对一大群具有典型特征的个体进行了基因分型,这些个体接受了自闭症谱系特征(自闭症商数,AQ)和分裂型谱系特征(分裂型人格问卷,SPQ-BR)评分,检测了非同义功能性SHANK3单核苷酸多态性rs9616915。男性而非女性在与言语和语言相关的SPQ-BR子量表“怪异言语”上表现出显著的基因型效应。这些发现,结合动物模型研究显示SHANK3突变对发声和听觉的影响,以及表明费伦-麦克德米德综合征存在严重语言改变和与言语相关的白质束异常的研究,表明SHANK3对人类语言和言语的发展及表达有不同影响。对携带rs9616915不同基因型的典型个体进行成像遗传学和言语-语言研究,应该能为SHANK3突变个体和费伦-麦克德米德综合征患者言语和语言改变的神经学和心理学基础提供新的见解。