Arruda W O, Torres L F, Lombes A, DiMauro S, Cardoso B A, Teive H A, De Paola D, Seixas R R
Hospital Nossa Senhora das Graças and Hospital de Clínicas, Universidade Federal do Paraná.
Arq Neuropsiquiatr. 1990 Mar;48(1):32-43. doi: 10.1590/s0004-282x1990000100006.
The authors describe a family (mother, son and two daughters) with mitochondrial myopathy. The mother was asymptomatic. Two daughters had lactic acidosis and myoclonic epilepsy, mild dementia, ataxia, weakness and sensory neuropathy. The son suffered one acute hemiplegic episode due to an ischemic infarct in the right temporal region. All the patients studied had hypertension. EEG disclosed photomyoclonic response in the proband patient. Muscle biopsy disclosed ragged-red fibers and abnormal mitochondria by electron microscopy. Biochemical analysis showed a defect of cytochrome C oxidase in mitochondria isolated from skeletal muscle. Several clinical and genetic aspects of the mitochondrial encephalomyopathies are discussed.
作者描述了一个患有线粒体肌病的家庭(母亲、儿子和两个女儿)。母亲无症状。两个女儿患有乳酸性酸中毒和肌阵挛性癫痫、轻度痴呆、共济失调、肌无力和感觉神经病变。儿子因右侧颞区缺血性梗死经历了一次急性偏瘫发作。所有接受研究的患者都患有高血压。脑电图显示先证者有光肌阵挛反应。肌肉活检显示有破碎红纤维,电子显微镜下可见线粒体异常。生化分析表明,从骨骼肌分离出的线粒体中细胞色素C氧化酶存在缺陷。文中讨论了线粒体脑肌病的几个临床和遗传学方面的问题。