Hasan Arı, Ayberk Türkyılmaz, Ayşe Burcu Doğan Arı, Hakan Kardeş, Abdullah Sezer, Emine Ayça Cimbek, Şenay Savaş Erdeve, Gülay Karagüzel
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引用次数: 0
摘要
智力发育障碍,常染色体隐性遗传60 (MRT60, #617432)是一种罕见的遗传性疾病,以小头畸形、智力残疾、生长迟缓、癫痫发作和中枢神经系统异常为特征。这种疾病是由TATA盒子结合蛋白相关因子基因(TAF13)基因的双等位变异引起的。迄今为止,文献中仅报道了4例MRT60患者。在这项研究中,提出了两名新患者,表现出相似的表型特征,包括小头畸形、智力残疾和明显的生长迟缓。全外显子组分析显示TAF13基因存在致病性变异(c.119T> a . p.Met40Lys)。46,xy性发育障碍仅存在于当前患者中,是这种罕见疾病的新发现。由于TAF13在转录调节中发挥作用,因此它被认为可能导致性腺功能障碍。为了更好地了解这种疾病,有必要进行全面的功能研究,从而更深入地了解潜在的机制。
Two New Families With TAF13 Variant Presenting With Syndromic 46,XY Disorder of Sex Development: Expanding the Clinical Phenotype.
Intellectual developmental disorder, autosomal recessive 60 (MRT60, #617432) is an ultrarare genetic disorder characterized by microcephaly, intellectual disability, growth retardation, seizure, and central nervous system abnormalities. The disease is caused by biallelic variants in the TATA box-binding protein-associated factor gene (TAF13) gene. To date, only four patients with MRT60 have been reported in the literature. In this study, two new patients were presented, exhibiting similar phenotypic features including microcephaly, intellectual disability, and prominent growth retardation. Whole exome analysis revealed a pathogenic variant (c.119T>A p.Met40Lys) in the TAF13 gene. The 46,XY disorder of sex development was only present in the current patients and is a new finding for this ultrarare disorder. Since TAF13 plays a role in transcriptional regulation, it is believed to potentially cause gonadal dysfunction. To obtain a better understanding of this disorder, it is essential to conduct comprehensive functional studies that can provide deeper insights into the underlying mechanisms.
期刊介绍:
The American Journal of Medical Genetics - Part A (AJMG) gives you continuous coverage of all biological and medical aspects of genetic disorders and birth defects, as well as in-depth documentation of phenotype analysis within the current context of genotype/phenotype correlations. In addition to Part A , AJMG also publishes two other parts:
Part B: Neuropsychiatric Genetics , covering experimental and clinical investigations of the genetic mechanisms underlying neurologic and psychiatric disorders.
Part C: Seminars in Medical Genetics , guest-edited collections of thematic reviews of topical interest to the readership of AJMG .