Santorelli F M, Shanske S, Macaya A, DeVivo D C, DiMauro S
H. Houston Merritt Clinical Research Center for Muscular Dystrophy and Related Diseases, Columbia-Presbyterian Medical Center, New York, NY.
Ann Neurol. 1993 Dec;34(6):827-34. doi: 10.1002/ana.410340612.
Twelve patients with Leigh's syndrome from 10 families harbored a T > G point mutation at nt 8993 of mtDNA. This mutation, initially associated with neurogenic weakness, ataxia, and retinitis pigmentosa, was later found to result in the Leigh phenotype when present in a high percentage. In our patients, the mutation was heteroplasmic, maternally inherited, and appeared to segregate rapidly within the pedigrees. Quantitative analysis revealed a good correlation between percentage of mutant mitochondrial genomes and severity of the clinical phenotype. The mutation was not found in > 200 patients with other mitochondrial encephalomyopathies or in controls. Mitochondrial enzyme activities were normal in all but 1 patient, and there were no ragged-red fibers in the muscle biopsy. Lactic acidosis was present in 92% of patients. Our findings suggest that the mtDNA nt 8993 mutation is a relatively common cause of Leigh's syndrome.
来自10个家庭的12例Leigh综合征患者线粒体DNA(mtDNA)的8993位核苷酸发生了T>G点突变。该突变最初与神经源性肌无力、共济失调和色素性视网膜炎相关,后来发现当以高比例存在时会导致Leigh综合征表型。在我们的患者中,该突变是异质性的,通过母系遗传,并且似乎在系谱中迅速分离。定量分析显示突变型线粒体基因组的比例与临床表型的严重程度之间存在良好的相关性。在200多名患有其他线粒体脑肌病的患者或对照组中未发现该突变。除1例患者外,所有患者的线粒体酶活性均正常,肌肉活检中未发现破碎红纤维。92%的患者存在乳酸性酸中毒。我们的研究结果表明,mtDNA 8993位核苷酸突变是Leigh综合征相对常见的病因。