Smeitink J A, Sengers R C, Trijbels J M, Ruitenbeek W, Daniëls O, Stadhouders A M, Kock-Jansen M J
Institute of Paediatrics, University of Nijmegen, The Netherlands.
Eur J Pediatr. 1989 Jun;148(7):656-9. doi: 10.1007/BF00441527.
Three patients suffering from the neonatal form of a syndrome characterized by congenital cataract, hypertrophic cardiomyopathy, and mitochondrial myopathy are described. The patients died at 7, 10 and 18 days, respectively from cardiorespiratory failure. Mitochondrial abnormalities were observed in the heart and skeletal muscle. Despite the presence of a severe lactic acidaemia pointing to a disturbed pyruvate oxidation rate in vivo, a normal pyruvate oxidation rate was demonstrated in skeletal muscle homogenates. The activities of several enzymes of the mitochondrial respiratory chain appeared to be normal, indicating an intact respiratory chain. A myoglobinopenia could be excluded. The activities of some mitochondrial enzymes and the concentration of myoglobin increase with age.
本文描述了三名患有以先天性白内障、肥厚性心肌病和线粒体肌病为特征的新生儿综合征的患者。这些患者分别在7天、10天和18天时死于心肺衰竭。在心脏和骨骼肌中观察到线粒体异常。尽管存在严重的乳酸血症,表明体内丙酮酸氧化速率紊乱,但在骨骼肌匀浆中丙酮酸氧化速率正常。线粒体呼吸链的几种酶的活性似乎正常,表明呼吸链完整。可排除肌红蛋白减少症。一些线粒体酶的活性和肌红蛋白的浓度随年龄增加。