Taylor Robert W, Schaefer Andrew M, McFarland Robert, Maddison Paul, Turnbull Douglass M
Department of Neurology, The Medical School, University of Newcastle upon Tyne, Framlington Place, NE2 4HH, Newcastle upon Tyne, UK.
Neuromuscul Disord. 2002 Oct;12(7-8):659-664. doi: 10.1016/s0960-8966(02)00026-3.
We describe a novel mutation in the mitochondrial tRNA(Ile) gene, an A to G transition at nucleotide position 4267, in a 37-year-old woman with myopathy, ataxia and sensorineural hearing loss. The A4267G mutation was heteroplasmic in several of the proband's tissues and single fibre analysis revealed significantly higher levels of mutated mitochondrial DNA in cytochrome c oxidase-deficient fibres than cytochrome c oxidase-positive fibres. It is predicted to disrupt a highly conserved base pair within the aminoacyl acceptor stem of the tRNA causing functional impairment, and as such fulfils all the accepted criteria for pathogenicity. Moreover, we were unable to detect the A4267G mutation in lymphocytes, buccal epithelia and hair of the patient's mother and two siblings, implying that the A4267G transition represents a sporadic, germline mutation.
我们描述了一名37岁患有肌病、共济失调和感音神经性听力损失的女性线粒体tRNA(Ile)基因中的一种新突变,即核苷酸位置4267处的A到G转换。A4267G突变在该先证者的多个组织中呈异质性,单纤维分析显示,细胞色素c氧化酶缺陷纤维中的突变线粒体DNA水平明显高于细胞色素c氧化酶阳性纤维。预计该突变会破坏tRNA氨酰基受体茎内一个高度保守的碱基对,导致功能受损,因此符合所有公认的致病性标准。此外,我们在患者母亲和两个兄弟姐妹的淋巴细胞、颊上皮细胞和头发中未检测到A4267G突变,这意味着A4267G转换代表一种散发性的种系突变。