Koga Y, Nonaka I, Kobayashi M, Tojyo M, Nihei K
Division of Ultrastructural Research, National Institute of Neuroscience, Tokyo, Japan.
Ann Neurol. 1988 Dec;24(6):749-56. doi: 10.1002/ana.410240609.
Thirteen of 15 patients with complex I deficiency had the multisystemic form, with strokelike episodes and other symptoms that fulfilled the diagnostic requirements for MELAS (mitochondrial myopathy, encephalopathy, lactic acidosis, and strokelike episodes), and 2 had only muscle fatigability and weakness, having the purely myopathic form. In the multisystemic form, 12 patients had ragged-red fibers. All multisystemic patients had myopathic histochemical abnormalities that consisted of mild to moderate variation in fiber size, disorganized intermyofibrillar networks, type 2 fiber atrophy, and an increased number of type 2C fibers. Five of 13 multisystemic patients had decreased cytochrome c oxidase (CCO) activity in extrafusal fibers, with sparing of intrafusal muscle fibers. In the myopathic form, pathological findings were similar to those in the multisystemic form. In addition to complex I and NADH dehydrogenase activities being decreased, the CCO activity was significantly decreased (less than 50% of control value) in 8 patients, especially when the disease was in its advanced stages, suggesting that CCO enzyme might be secondarily affected as the disease progresses.
15例复合体I缺乏患者中有13例表现为多系统形式,伴有类卒中发作及其他符合线粒体肌病、脑病、乳酸酸中毒和类卒中发作(MELAS)诊断标准的症状,2例仅有肌肉疲劳和无力,为单纯肌病形式。在多系统形式中,12例患者有破碎红纤维。所有多系统患者均有肌病组织化学异常,包括纤维大小轻度至中度变异、肌原纤维间网络紊乱、2型纤维萎缩以及2C型纤维数量增加。13例多系统患者中有5例的梭外肌纤维细胞色素c氧化酶(CCO)活性降低,而梭内肌纤维不受影响。在肌病形式中,病理表现与多系统形式相似。除了复合体I和NADH脱氢酶活性降低外,8例患者的CCO活性显著降低(低于对照值的50%),尤其是在疾病晚期,这表明随着疾病进展,CCO酶可能受到继发性影响。