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迟发性视神经萎缩和共济失调中线粒体呼吸链复合体II缺乏。

Deficiency of complex II of the mitochondrial respiratory chain in late-onset optic atrophy and ataxia.

作者信息

Taylor R W, Birch-Machin M A, Schaefer J, Taylor L, Shakir R, Ackrell B A, Cochran B, Bindoff L A, Jackson M J, Griffiths P, Turnbull D M

机构信息

Departments of Neurology and Ophthalmology, University of Newcastle upon Tyne, U.K.

出版信息

Ann Neurol. 1996 Feb;39(2):224-32. doi: 10.1002/ana.410390212.

Abstract

Defects of the mitochondrial respiratory chain are increasingly being recognized as an important cause of neurological disease in humans. In many of these patients, the biochemical defect results from an abnormality of the mitochondrial genome. Respiratory chain defects involving complex II, which is entirely encoded by the nuclear genome, are comparatively rare. We report the clinical and biochemical findings in 2 elderly sisters who presented with late-onset neurodegenerative disease. In both patients, a partial deficiency of complex II (approximately 50% of control values) was shown to be present in mitochondria from muscle and platelets. The enzyme defect was not expressed in cultured skin fibroblasts or immortalized lymphocytes. There was an overexpression of the 70-kd flavoprotein subunit in muscle mitochondria from both patients, although we showed that this subunit is present in normal amounts in mitochondrial membranes. Our studies highlight the diversity of the clinical presentation of respiratory chain disease and that complex II deficiency should enter the differential diagnosis of certain patients with late-onset neurodegenerative disease.

摘要

线粒体呼吸链缺陷日益被认为是人类神经疾病的一个重要病因。在许多这类患者中,生化缺陷是由线粒体基因组异常导致的。涉及复合体II的呼吸链缺陷相对少见,因为复合体II完全由核基因组编码。我们报告了2例患有迟发性神经退行性疾病的老年姐妹的临床和生化检查结果。在这两名患者中,肌肉和血小板线粒体中均显示存在复合体II部分缺陷(约为对照值的50%)。酶缺陷在培养的皮肤成纤维细胞或永生化淋巴细胞中未表现出来。两名患者的肌肉线粒体中70-kd黄素蛋白亚基均有过表达,尽管我们发现该亚基在线粒体膜中的含量正常。我们的研究突出了呼吸链疾病临床表现的多样性,以及复合体II缺陷应纳入某些迟发性神经退行性疾病患者的鉴别诊断。

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