Campos Y, Martín M A, García-Silva T, del Hoyo P, Rubio J C, Castro-Gago M, García-Peñas J, Casas J, Cabello A, Ricoy J R, Arenas J
Centro de Investigación, Hospital Universitario 12 de Octubre, Madrid, Spain.
Neuromuscul Disord. 1998 Dec;8(8):568-73. doi: 10.1016/s0960-8966(98)00080-7.
We studied 10 patients with a variable degree of mtDNA depletion in muscle. Seven patients showed a clear-cut myopathic pattern, while the three remaining had brain involvement. There was no relationship between age at onset and relative mtDNA copy number in muscle, but we found an apparent correlation between clinical severity and degree of muscle mtDNA depletion. Muscle morphology showed that mtDNA depletion was associated with mitochondrial proliferation and cytochrome c oxidase negative fibers. Biochemical studies revealed single or combined defects of mtDNA-dependent respiratory chain complexes. Our data indicate that patients with mtDNA depletion may have a more variable age at onset and clinical evolution and wider phenotype than previously thought. The diagnosis of this condition, so far regarded as rare, may have been overlooked to some extent.
我们研究了10例肌肉中线粒体DNA(mtDNA)有不同程度缺失的患者。7例患者呈现出明确的肌病模式,而其余3例有脑部受累情况。发病年龄与肌肉中相对mtDNA拷贝数之间没有关联,但我们发现临床严重程度与肌肉mtDNA缺失程度之间存在明显相关性。肌肉形态学显示,mtDNA缺失与线粒体增殖及细胞色素c氧化酶阴性纤维有关。生化研究揭示了mtDNA依赖性呼吸链复合物存在单一或联合缺陷。我们的数据表明,mtDNA缺失患者的发病年龄和临床进展可能比之前认为的更具多样性,且表型更广泛。这种迄今为止被认为罕见的疾病诊断在一定程度上可能被忽视了。