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第二位非典型线粒体骨骼肌受累的 MNGIE 患者。

A second MNGIE patient without typical mitochondrial skeletal muscle involvement.

机构信息

Department of Neurological, Neurosurgical and Behavioural Sciences, University of Siena, Viale Bracci 2, 53100, Siena, Italy.

出版信息

Neurol Sci. 2010 Aug;31(4):491-4. doi: 10.1007/s10072-010-0225-5. Epub 2010 Mar 16.

Abstract

Mitochondrial neurogastrointestinal encephalomyopathy (MNGIE) is an autosomal recessive disease caused by mutations in the gene encoding thymidine phosphorylase (TYMP). Clinically, MNGIE is characterized by gastrointestinal dysmotility, cachexia, ptosis, ophthalmoparesis, peripheral neuropathy and leukoencephalopathy. Most MNGIE patients have signs of mitochondrial dysfunction in skeletal muscle at morphological and enzyme level, as well as mitochondrial DNA depletion, multiple deletions and point mutations. A case without mitochondrial skeletal muscle involvement and with a TYMP splice-acceptor site mutation (c. 215-1 G>C) has been reported. Here, we describe an Italian patient with the same mutation and without mitochondrial skeletal muscle involvement, suggesting a possible genotype-phenotype correlation.

摘要

线粒体神经胃肠型脑肌病(MNGIE)是一种常染色体隐性疾病,由编码胸苷磷酸化酶(TYMP)的基因突变引起。临床上,MNGIE 的特征是胃肠道动力障碍、恶病质、眼睑下垂、眼肌麻痹、周围神经病和脑白质病。大多数 MNGIE 患者在形态学和酶水平上都有骨骼肌线粒体功能障碍的迹象,以及线粒体 DNA 耗竭、多处缺失和点突变。已经报道了一例没有线粒体骨骼肌受累和 TYMP 剪接受位突变(c.215-1G>C)的病例。在这里,我们描述了一位具有相同突变且没有线粒体骨骼肌受累的意大利患者,提示可能存在基因型-表型相关性。

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