Monden Yukifumi, Mori Masato, Kuwajima Mari, Goto Tamako, Yamagata Takanori, Momoi Mariko Y
Department of Pediatrics, Jichi Medical University, 3311-1 Yakushiji, Shimotsuke, Tochigi, Japan.
Brain Dev. 2013 Jun;35(6):582-5. doi: 10.1016/j.braindev.2012.08.006. Epub 2012 Sep 13.
We report the case of a boy with myoclonic epilepsy with ragged-red fibers (MERRF) who had astatic seizures since 2 years of age and later developed ataxia, absence seizures, and myoclonus. Almost homoplasmic A8344G mutation of mitochondrial DNA (m.8344A>G mutation) was detected in lymphocytes. He developed late-onset Leigh syndrome (LS) when he contracted pneumonia at 6 years. He developed bulbar palsy and deep coma. MRI demonstrated lesions in the brainstem, basal ganglia, and cerebral cortex. Three similar cases have been reported; two carried the almost-homoplasmic m.8344A>G mutation in muscle tissue. These suggested that almost homoplastic m.8344A>G mutation developed clinical phenotype of MERRF in the early stage and late-onset Leigh syndrome in the late course of the disease.
我们报告了一例患有肌阵挛性癫痫伴破碎红纤维(MERRF)的男孩病例,该男孩自2岁起出现失张力发作,随后发展为共济失调、失神发作和肌阵挛。在淋巴细胞中检测到线粒体DNA几乎完全同质的A8344G突变(m.8344A>G突变)。他6岁患肺炎时发展为迟发性 Leigh 综合征(LS)。他出现了延髓麻痹和深度昏迷。MRI显示脑干、基底神经节和大脑皮质有病变。已报道了另外3例类似病例;其中2例在肌肉组织中携带几乎完全同质的m.8344A>G突变。这些提示几乎完全同质的m.8344A>G突变在疾病早期表现为MERRF的临床表型,而在疾病后期发展为迟发性Leigh综合征。