Hiruta Y, Muto M, Ichihara T, Uruga K, Mochizuki M, Wachi E, Miyabayashi S, Mayumi F, Adachi K, Toshima H
Department of Cardiology, Internal Medicine, Iwaki Kyoritsu General Hospital.
Kokyu To Junkan. 1993 Mar;41(3):281-6.
We report an autopsy case of a 19 year-old man with MELAS (mitochondrial myopathy, encephalopathy, lactic acidosis, and stroke-like episodes) a subgroup of mitochondrial encephalomyopathy presenting cardiomyopathy. He had repeatedly suffered from transient unconsciousness, hemiplegia, hemianopsia and convulsion attacks since the age of 9, and he died of severe congestive heart failure. In laboratory findings, blood lactate and pyruvate were markedly increased. Skeletal muscle biopsy demonstrated numerously scattered ragged-red fibers with modified Gomori's trichrome staining. Enzymatic activities of the mitochondrial respiratory chain showed a marked decrease of NADH cytochrome c reductase (complex I). In postmortem examination, the heart was 310g in weight and had right ventricular dilatation. Microscopically, degenerated and scattered myocardial cells (ragged-red fibers), interstitial edema and microvascular hyperplasia were demonstrated in the myocardium. Under the electron microscope, abnormal mitochondria proliferated and myofibrils were unusually sparse. Immunohistochemical studies with specific antibodies against the mitochondrial electron transfer enzyme subunits revealed a reduction of immunoreactive materials for complex I in the myocardium. These results suggested the relationship of myocardial disorders and decreased activity of complex I in electron transfer enzymes in this patient.
我们报告一例19岁男性线粒体脑肌病伴乳酸性酸中毒及卒中样发作(MELAS)患者的尸检病例,MELAS是线粒体脑肌病的一个亚组,伴有心肌病。自9岁起,他反复出现短暂性意识丧失、偏瘫、偏盲和惊厥发作,最终死于严重的充血性心力衰竭。实验室检查发现,血乳酸和丙酮酸显著升高。骨骼肌活检显示,改良Gomori三色染色可见大量散在的破碎红纤维。线粒体呼吸链的酶活性显示NADH细胞色素c还原酶(复合体I)显著降低。尸检时,心脏重量为310克,右心室扩张。显微镜下,心肌可见变性和散在的心肌细胞(破碎红纤维)、间质水肿和微血管增生。电子显微镜下,异常线粒体增生,肌原纤维异常稀疏。针对线粒体电子传递酶亚基的特异性抗体进行免疫组织化学研究显示,心肌中复合体I的免疫反应物质减少。这些结果提示该患者心肌病变与电子传递酶中复合体I活性降低之间存在关联。