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由于 CHD8 基因的变异,自闭症谱系障碍的表型和基因型数据得到扩展。

Expansion of phenotypic and genotypic data in autism spectrum disorders due to variants in the CHD8 gene.

机构信息

Veltischev Research and Clinical Institute for Pediatrics and Pediatric Surgery of the Pirogov Russian National Research Medical University of the Ministry of Health of the Russian Federation, Moscow, Russia.

出版信息

Neurogenetics. 2024 Nov 22;26(1):4. doi: 10.1007/s10048-024-00781-9.

Abstract

Autism spectrum disorders are a group of the most common disorders of neuropsychiatric development, characterized by difficulties in social interaction and adherence to stereotypic behavioral patterns. This group of conditions frequently co-occurs with intellectual disability, epilepsy, attention-deficit hyperactivity disorder, connective tissue disorders and others. Among the most common molecular-genetic causes of autism spectrum disorders are pathogenic variants in the CHD8 gene. CHD8 codes for chromodomain-helicase-DNA-binding protein 8 - a chromatin remodeler that regulates cellular proliferation and brain development in embryogenesis. 6 children and 1 adult (mother of 1 of the children) and were found to have clinically significant variants in CHD8 on whole genome sequencing (3 children and 1 adult had likely pathogenic variants, 3 children- variants of unknown significance). Their phenotype consisted of autism spectrum disorders, developmental delay, ataxia, overgrowth and other signs typically observed in patients with pathogenic variants in CHD8, as well as common comorbidities of autism spectrum disorders, such as attention-deficit hyperactivity disorder and connective tissue disorders. Additionally, 4 patients had hepatomegaly and 2- hyperbilirubinemia (1 had both) - clinical features have not been previously associated with pathogenic variants in CHD8. 2 patients also presented with cardiovascular abnormalities, primarily arrythmias and, in 1 case, cardiomyopathy- also uncharacteristic of patients with pathogenic variants in CHD8. Further research is required to determine the mechanisms underlying the abovementioned clinical features, which are likely carried out through complex interactions between CHD8 and other regulatory proteins.

摘要

自闭症谱系障碍是一组最常见的神经精神发育障碍,其特征是社交互动困难和坚持刻板的行为模式。这组疾病常与智力障碍、癫痫、注意力缺陷多动障碍、结缔组织疾病等并存。自闭症谱系障碍最常见的分子遗传病因之一是 CHD8 基因的致病性变异。CHD8 编码染色质调节蛋白 8 的色氨酸-螺旋-环-螺旋结构域-螺旋酶,该蛋白在胚胎发生中调节细胞增殖和大脑发育。在全基因组测序中发现 6 名儿童和 1 名成年人(其中 1 名儿童的母亲)存在 CHD8 中具有临床意义的变异(3 名儿童和 1 名成年人具有可能的致病性变异,3 名儿童为意义不明的变异)。他们的表型包括自闭症谱系障碍、发育迟缓、共济失调、过度生长和其他通常在 CHD8 致病性变异患者中观察到的体征,以及自闭症谱系障碍的常见共病,如注意力缺陷多动障碍和结缔组织疾病。此外,4 名患者有肝肿大,2 名-高胆红素血症(1 名两者兼有)-这些临床特征以前与 CHD8 中的致病性变异无关。2 名患者还出现心血管异常,主要是心律失常,在 1 例中出现心肌病-这也不符合 CHD8 致病性变异患者的特征。需要进一步研究以确定上述临床特征的潜在机制,这些特征可能是通过 CHD8 与其他调节蛋白之间的复杂相互作用来实现的。

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