Blakely Emma, He Langping, Gardner Julie L, Hudson Gavin, Walter John, Hughes Imelda, Turnbull Douglass M, Taylor Robert W
Mitochondrial Research Group, The Medical School, Newcastle University, Newcastle Upon Tyne NE2 4HH, UK.
Neuromuscul Disord. 2008 Jul;18(7):557-60. doi: 10.1016/j.nmd.2008.04.014. Epub 2008 May 27.
Mitochondrial DNA depletion syndromes are a heterogeneous group of childhood neurological disorders characterised by a quantitative abnormality of mitochondrial DNA. We describe two siblings who presented at 8 months and 14 months with myopathy, which rapidly progressed and resulted in death by respiratory failure at age 14 and 18 months, respectively. Muscle biopsy revealed marked respiratory chain defects, with real-time PCR confirming a dramatic depletion of mitochondrial DNA. Sequencing of the thymidine kinase 2 (TK2) gene revealed two, novel heterozygous mutations (p.Q87X and p.N100S) with parental DNA analysis confirming the transmission of mutated alleles.
线粒体DNA耗竭综合征是一组异质性的儿童神经障碍,其特征为线粒体DNA的数量异常。我们描述了两名分别在8个月和14个月时出现肌病的兄弟姐妹,病情迅速进展,分别于14个月和18个月时因呼吸衰竭死亡。肌肉活检显示明显的呼吸链缺陷,实时PCR证实线粒体DNA显著耗竭。胸苷激酶2(TK2)基因测序发现两个新的杂合突变(p.Q87X和p.N100S),对父母的DNA分析证实了突变等位基因的传递。