Suomalainen A, Ciafaloni E, Koga Y, Peltonen L, DiMauro S, Schon E A
Department of Neurology, Columbia University, New York, NY.
J Neurol Sci. 1992 Sep;111(2):222-6. doi: 10.1016/0022-510x(92)90074-u.
Myoclonus epilepsy with ragged-red fibers (MERRF) has been shown to be associated with a specific point mutation at the nucleotide 8344 in the tRNA(Lys) gene of mitochondrial DNA (mtDNA). We screened 6 patients with clinically diagnosed MERRF and 1 patient with ocular myopathy for point mutations in the tRNA(Lys) gene, using single strand conformation polymorphism (SSCP) analysis, which can detect even a 1-basepair difference between 2 DNA sequences. Using SSCP and consequent DNA sequencing, we identified the known MERRF mutation in 4 out of 6 MERRF patients, as well as in 1 patient with a new clinical phenotype associated with this mutation: progressive external ophthalmoplegia, muscle weakness and a lipoma, but no myoclonus or epilepsy. Two of the patients with clinical MERRF had neither the MERRF-mutation nor any other mutations in the tRNA(Lys) gene. Using SSCP analysis, we also detected a new polymorphism in 1 patient. Thus, SSCP analysis can be applied to search effectively and rapidly for point mutations or polymorphisms in mitochondrial DNA.
肌阵挛性癫痫伴破碎红纤维病(MERRF)已被证明与线粒体DNA(mtDNA)的tRNA(Lys)基因中第8344位核苷酸的特定点突变有关。我们使用单链构象多态性(SSCP)分析,对6例临床诊断为MERRF的患者和1例眼肌病患者进行了tRNA(Lys)基因点突变筛查,该分析甚至可以检测到两条DNA序列之间1个碱基对的差异。通过SSCP及后续的DNA测序,我们在6例MERRF患者中的4例以及1例具有与该突变相关的新临床表型(进行性眼外肌麻痹、肌肉无力和脂肪瘤,但无肌阵挛或癫痫)的患者中鉴定出了已知的MERRF突变。2例临床诊断为MERRF的患者既没有MERRF突变,tRNA(Lys)基因中也没有任何其他突变。通过SSCP分析,我们还在1例患者中检测到了一种新的多态性。因此,SSCP分析可用于有效、快速地搜索线粒体DNA中的点突变或多态性。