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伴有破碎红纤维的肌阵挛性癫痫的单肌纤维分析

Single muscle fiber analysis of myoclonus epilepsy with ragged-red fibers.

作者信息

Mita S, Tokunaga M, Uyama E, Kumamoto T, Uekawa K, Uchino M

机构信息

Department of Neurology, Kumamoto University School of Medicine, Japan.

出版信息

Muscle Nerve. 1998 Apr;21(4):490-7. doi: 10.1002/(sici)1097-4598(199804)21:4<490::aid-mus7>3.0.co;2-4.

Abstract

We examined two muscle biopsy specimens from a proband and her mother with myoclonus epilepsy with ragged-red fibers (MERRF), both obtained at an interval of about 10 years, using histochemistry, in situ hybridization, and single-fiber polymerase chain reaction. Total (wild-type and mutant) mitochondrial DNAs (mtDNAs) were greatly increased in ragged-red fibers (RRF) over non-RRF in all muscle specimens analyzed. The proportion of mutant mtDNA was also significantly higher in RRF than in non-RRF. By comparing the first and second muscle biopsied specimens in each patient, we found that while the proportion of RRF, cytochrome coxidase deficient fibers, and mutant DNA in muscle changed over a 10-year period, the proportion of wild-type and mutant mtDNAs in RRF and in non-RRF was similar between the two specimens. These results suggest that the ratio of wild-type to mutant mtDNAs in RRF and non-RRF in MERRF is at a steady state level in each muscle fiber, without replicative advantage of mutant mtDNA.

摘要

我们使用组织化学、原位杂交和单纤维聚合酶链反应,对一名患有肌阵挛性癫痫伴破碎红纤维(MERRF)的先证者及其母亲的两份肌肉活检标本进行了检查,两份标本采集间隔约10年。在所有分析的肌肉标本中,破碎红纤维(RRF)中的总(野生型和突变型)线粒体DNA(mtDNA)相较于非RRF显著增加。RRF中突变型mtDNA的比例也显著高于非RRF。通过比较每位患者的首次和第二次肌肉活检标本,我们发现,虽然在10年期间肌肉中RRF、细胞色素c氧化酶缺陷纤维和突变DNA的比例发生了变化,但两份标本中RRF和非RRF中野生型和突变型mtDNA的比例相似。这些结果表明,MERRF中RRF和非RRF中野生型与突变型mtDNA的比例在每条肌纤维中处于稳定状态,突变型mtDNA没有复制优势。

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