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线粒体神经胃肠性脑肌病中TYMP基因的新突变:病例系列及文献综述

Novel Mutations of the TYMP Gene in Mitochondrial Neurogastrointestinal Encephalomyopathy: Case Series and Literature Review.

作者信息

Mojtabavi Helia, Fatehi Farzad, Shahkarami Sepideh, Rezaei Nima, Nafissi Shahriar

机构信息

School of Medicine, Tehran University of Medical Sciences, Tehran, Iran.

Systematic Review and Meta-Analysis Expert Group (SRMEG), Universal Scientific Education and Research Network (USERN), Tehran, Iran.

出版信息

J Mol Neurosci. 2021 Dec;71(12):2526-2533. doi: 10.1007/s12031-021-01822-w. Epub 2021 Apr 6.

Abstract

Mitochondrial neurogastrointestinal encephalomyopathy (MNGIE) is a multi-system disorder caused by several homozygous or compound heterozygous mutations, mostly in the nuclear gene of TYMP. Our current knowledge on the underlying pathology of the disease is derived through the study of about 200 cases of different ethnicities. Clinical presentations include severe cachexia, weakness, ptosis, diplopia, abdominal cramps or digestive tract disorders, hearing impairment, and paresthesia.Herein, we aim to present five novel mutations of the nuclear gene of TYMP in six Iranian patients diagnosed with MNGIE. In our population, age at the time of diagnosis was 18 to 49 years, while the onset of the symptoms varied from 13 to 20 years. We detected two pathogenic non-frameshift nonsense premature stop codon mutations (c.1013C > A, and c.130C > T), one variant of uncertain significance (VUS) non-frameshift missense mutation (c.345G > T), one likely pathogenic frameshift insertion (c.801_802insCGCG), and one likely benign homozygous non-frameshift deletion (c.1176_1187del) from two siblings. Our findings also confirm the autosomal recessive inheritance pattern of MNGIE in the Iranian population. The lack of knowledge in the area of nuclear gene-modifier genes shadows the genotype-phenotype relationships of MNGIE.

摘要

线粒体神经胃肠性脑肌病(MNGIE)是一种多系统疾病,由多个纯合或复合杂合突变引起,这些突变大多位于TYMP核基因中。我们目前对该疾病潜在病理的认识来自于对约200例不同种族患者的研究。临床表现包括严重恶病质、虚弱、上睑下垂、复视、腹部绞痛或消化道疾病、听力障碍和感觉异常。在此,我们旨在介绍6例被诊断为MNGIE的伊朗患者中TYMP核基因的5种新突变。在我们的研究人群中,诊断时的年龄为18至49岁,而症状出现的年龄在13至20岁之间。我们检测到两个致病性非移码无义提前终止密码子突变(c.1013C>A和c.130C>T)、一个意义未明的变异(VUS)非移码错义突变(c.345G>T)、一个可能致病的移码插入(c.801_802insCGCG)以及来自两名同胞的一个可能良性的纯合非移码缺失(c.1176_1187del)。我们的研究结果也证实了MNGIE在伊朗人群中的常染色体隐性遗传模式。核基因修饰基因领域知识的缺乏使MNGIE的基因型 - 表型关系变得模糊。

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