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线粒体肌阵挛性癫痫伴破碎红纤维(MERRF)突变A8344G患者的不典型表现。

Unusual presentations of patients with the mitochondrial MERRF mutation A8344G.

作者信息

Wiedemann Falk R, Bartels Claudius, Kirches Elmar, Mawrin Christian, Wallesch Claus-W

机构信息

Klinik für Neurologie, Otto-von-Guericke Universität, D-39120 Magdeburg, Germany.

出版信息

Clin Neurol Neurosurg. 2008 Sep;110(8):859-63. doi: 10.1016/j.clineuro.2008.06.010. Epub 2008 Jul 26.

Abstract

MERRF is typically characterized by myoclonus, generalized seizures and ragged-red fibers in muscular biopsy. We report a family (harbouring the A8344G mutation) with a late onset of the disease and an uncommon clinical manifestation, including episodes of reversible respiratory failure, the presence of ophthalmoplegia, and the absence of seizures and myoclonus in most subjects. We conducted histochemical, biochemical and molecular genetic studies. Mutation analysis revealed that the level of mutated mitochondrial DNA (mtDNA) was above 80% in the skeletal muscle of all siblings. Nevertheless, one severely affected individual did neither present cytochrome c oxidase-negative fibers nor ragged-red fibers in the skeletal muscle biopsy. These data extend the phenotypic range associated with the MERRF syndrome. We suggest that the analysis of mtDNA could be of importance in many cases of unclear multisystem disorders in later life.

摘要

肌阵挛性癫痫伴破碎红纤维病(MERRF)的典型特征为肌阵挛、全身性癫痫发作以及肌肉活检可见破碎红纤维。我们报告了一个携带A8344G突变的家系,该家系疾病起病较晚且临床表现不常见,包括可逆性呼吸衰竭发作、存在眼肌麻痹,且大多数患者无癫痫发作和肌阵挛。我们进行了组织化学、生物化学和分子遗传学研究。突变分析显示,所有兄弟姐妹的骨骼肌中突变型线粒体DNA(mtDNA)水平均高于80%。然而,一名严重受累个体的骨骼肌活检既未发现细胞色素c氧化酶阴性纤维,也未发现破碎红纤维。这些数据扩展了与MERRF综合征相关的表型范围。我们认为,mtDNA分析在许多老年期不明原因的多系统疾病病例中可能具有重要意义。

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