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线粒体脑肌病重叠综合征的眼科临床病理研究

Ocular clinicopathologic study of the mitochondrial encephalomyopathy overlap syndromes.

作者信息

Chang T S, Johns D R, Walker D, de la Cruz Z, Maumence I H, Green W R

机构信息

Eye Pathology Laboratory, Wilmer Institute, Johns Hopkins Medical Institutions, Baltimore, Md.

出版信息

Arch Ophthalmol. 1993 Sep;111(9):1254-62. doi: 10.1001/archopht.1993.01090090106028.

Abstract

Recent advances in molecular genetics have led to a better understanding of mitochondrially inherited diseases. Mitochondrial encephalomyopathy overlap syndrome is one such group of diseases in which ocular abnormalities are frequently manifest. The authors describe the clinical, molecular genetic, and pathologic findings of two patients with the mitochondrial encephalomyopathy overlap syndrome. The patients shared a similar clinical course with features overlapping the three traditionally distinct clinical phenotypes (the Kearns-Sayre syndrome; the syndrome of mitochondrial encephalopathy, lactic acidosis, and stroke [MELAS], and the syndrome of myoclonus, epilepsy, and ragged red fibers [MERRF]). The patients had identical mitochondrial DNA mutations (at nucleotide position 3243) and had similar ultrastructural abnormalities, including abundant enlarged mitochondria with "whorled" and "tubular" cristae. These abnormal mitochondria appeared to be preferentially distributed in cells with high metabolic activity (retinal pigment epithelium, corneal endothelium, and extraocular muscles).

摘要

分子遗传学的最新进展使人们对线粒体遗传病有了更好的理解。线粒体脑肌病重叠综合征就是这样一组经常表现出眼部异常的疾病。作者描述了两名线粒体脑肌病重叠综合征患者的临床、分子遗传学和病理学发现。这两名患者有着相似的临床病程,其特征与三种传统上不同的临床表型(卡恩斯-塞尔综合征;线粒体脑肌病、乳酸酸中毒和卒中综合征[MELAS];肌阵挛、癫痫和破碎红纤维综合征[MERRF])重叠。这两名患者有相同的线粒体DNA突变(位于核苷酸位置3243),并且有相似的超微结构异常,包括大量增大的线粒体,其嵴呈“涡状”和“管状”。这些异常线粒体似乎优先分布于具有高代谢活性的细胞(视网膜色素上皮、角膜内皮和眼外肌)中。

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