Division of Neurology, Cincinnati Children's Hospital Medical Center, Cincinnati, OH 45229-3026, USA.
Neuromuscul Disord. 2009 Nov;19(11):784-7. doi: 10.1016/j.nmd.2009.08.002. Epub 2009 Sep 6.
The mitochondrial DNA depletion syndromes (MDS) are autosomal recessive disorders with a decreased mitochondrial DNA copy number. Mutations in thymidine kinase 2 (TK2) have been responsible for the myopathic form of MDS. We describe a child with congenital muscle weakness who had a progressive mitochondrial myopathy associated with extensive fibro-fatty replacement of myofibers resembling muscular dystrophy. MDS was suspected based upon findings in the initial muscle biopsy. Sequence analysis of the TK2 gene revealed two novel heterozygous mutations: the frame shift mutation, c.255_c.258delAGAA, and the heterozygous missense mutation, c.515G>A, (p.R172Q). This report extends the phenotype and genotype of TK2 defects.
线粒体 DNA 耗竭综合征(MDS)是一种线粒体 DNA 拷贝数减少的常染色体隐性疾病。胸苷激酶 2(TK2)的突变导致了 MDS 的肌病形式。我们描述了一名患有先天性肌肉无力的儿童,其患有进行性线粒体肌病,伴有肌纤维的广泛纤维脂肪替代,类似于肌肉营养不良症。基于最初的肌肉活检结果,怀疑 MDS。TK2 基因的序列分析显示了两个新的杂合突变:框移突变 c.255_c.258delAGAA 和杂合错义突变 c.515G>A,(p.R172Q)。本报告扩展了 TK2 缺陷的表型和基因型。