Campos Y, Garcia-Silva T, Barrionuevo C R, Cabello A, Muley R, Arenas J
Centro de Investigacion, Hospital 12 de Octubre, Madrid, Spain.
Pediatr Neurol. 1995 Jul;13(1):69-72. doi: 10.1016/0887-8994(95)00082-q.
Large-scale mitochondrial DNA deletion was found in a 5-year-old girl with mitochondrial encephalomyopathy, lactic acidosis, and stroke-like episodes (MELAS) and Fanconi's syndrome. Muscle biopsy disclosed ragged-red fibers and cytochrome c oxidase negative fibers. Respiratory chain studies were normal. Southern blot analysis demonstrated a 10.5-Kb heteroplasmic deletion in both muscle and blood. Deleted genomes represented 40% of total mitochondrial DNA in muscle and 63% in blood. There was no evidence of point mutations characteristic of MELAS. We suggest that not only patients with progressive external ophthalmoplegia syndromes, but also those with defined syndromes [e.g., MELAS or myoclonic epilepsy and ragged-red fibers (MERRF)] without characteristic point mutations, be screened for mitochondrial DNA deletions.
在一名患有线粒体脑肌病、乳酸酸中毒和卒中样发作(MELAS)及范科尼综合征的5岁女孩中发现了大规模线粒体DNA缺失。肌肉活检显示有破碎红纤维和细胞色素c氧化酶阴性纤维。呼吸链研究正常。Southern印迹分析表明肌肉和血液中均存在10.5-Kb的异质性缺失。缺失的基因组在肌肉中占线粒体DNA总量的40%,在血液中占63%。没有证据表明存在MELAS特征性的点突变。我们建议,不仅要对进行性眼外肌麻痹综合征患者进行线粒体DNA缺失筛查,还要对那些没有特征性点突变的明确综合征患者[如MELAS或肌阵挛性癫痫伴破碎红纤维(MERRF)]进行筛查。