自闭症谱系障碍的表型和基因型数据因 CHD8 基因变异而扩大。

IF 1.6 4区 医学 Q3 CLINICAL NEUROLOGY Neurogenetics Pub Date : 2024-11-22 DOI:10.1007/s10048-024-00781-9
Mariia A Parfenenko, Ilya S Dantsev, Sergei V Bochenkov, Rabiat G Kuramagomedova, Natalia V Vinogradova, Mariia P Afanaseva, Olga S Groznova, Victoria Iu Voinova
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引用次数: 0

摘要

自闭症谱系障碍是一组最常见的神经精神发育障碍,其特点是社交互动困难和行为模式刻板。这类疾病经常与智力障碍、癫痫、注意力缺陷多动障碍、结缔组织病等并发。自闭症谱系障碍最常见的分子遗传学病因是 CHD8 基因的致病变异。CHD8 编码染色质域-蒜酶-DNA 结合蛋白 8,这是一种染色质重塑因子,在胚胎发育过程中调节细胞增殖和大脑发育。研究人员对 6 名儿童和 1 名成人(其中 1 名儿童的母亲)进行了全基因组测序,发现他们的 CHD8 基因存在具有临床意义的变异(3 名儿童和 1 名成人可能存在致病变异,3 名儿童存在意义不明的变异)。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 CHD8 致病变体患者通常会出现的其他症状,以及自闭症谱系障碍的常见合并症,如注意力缺陷多动障碍和结缔组织疾病。此外,4名患者有肝肿大和2-高胆红素血症(1名患者同时有这两种症状),这些临床特征以前从未与CHD8致病变异相关联。2 名患者还伴有心血管异常,主要是心律失常,其中 1 例还伴有心肌病--这也是 CHD8 致病变体患者的特征。上述临床特征很可能是通过 CHD8 和其他调节蛋白之间复杂的相互作用产生的,因此需要进一步的研究来确定其机制。
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Expansion of phenotypic and genotypic data in autism spectrum disorders due to variants in the CHD8 gene.

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.

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来源期刊
Neurogenetics
Neurogenetics 医学-临床神经学
CiteScore
3.90
自引率
0.00%
发文量
24
审稿时长
6 months
期刊介绍: Neurogenetics publishes findings that contribute to a better understanding of the genetic basis of normal and abnormal function of the nervous system. Neurogenetic disorders are the main focus of the journal. Neurogenetics therefore includes findings in humans and other organisms that help understand neurological disease mechanisms and publishes papers from many different fields such as biophysics, cell biology, human genetics, neuroanatomy, neurochemistry, neurology, neuropathology, neurosurgery and psychiatry. All papers submitted to Neurogenetics should be of sufficient immediate importance to justify urgent publication. They should present new scientific results. Data merely confirming previously published findings are not acceptable.
期刊最新文献
Epigenetic dysregulation in glioblastoma: potential pathways to precision medicine. Understanding pathophysiology in fragile X syndrome: a comprehensive review. DHDDS-related epilepsy with hippocampal atrophy: a case report. Expansion of phenotypic and genotypic data in autism spectrum disorders due to variants in the CHD8 gene. Dystrophinopathy patient data as a guide to interpretation of pregestational female population screening for DMD gene variants.
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