Hayashi J, Ohta S, Kikuchi A, Takemitsu M, Goto Y, Nonaka I
Department of Biochemistry, Saitama Cancer Center Research Institute, Japan.
Proc Natl Acad Sci U S A. 1991 Dec 1;88(23):10614-8. doi: 10.1073/pnas.88.23.10614.
Mutant mitochondrial DNA with large-scale deletions (delta-mtDNA) has been frequently observed in patients with chronic progressive external ophthalmoplegia (CPEO), a subgroup of the mitochondrial encephalomyopathies. To exclude involvement of the nuclear genome in expression of the mitochondrial dysfunction characteristic of CPEO, we introduced the mtDNA of a CPEO patient into clonal mtDNA-less HeLa cells and isolated cybrid clones. Quantitation of delta-mtDNA in the cybrids revealed that delta-mtDNA was selectively propagated with higher levels of delta-mtDNA correlating with slower cellular growth rate. In these cybrid clones, translational complementation of the missing tRNAs occurred only when delta-mtDNA was less than 60% of the total mtDNA, whereas accumulation of delta-mtDNA to greater than 60% resulted in progressive inhibition of overall mitochondrial translation as well as reduction of cytochrome c oxidase activity throughout the organelle population. Because these cybrids shared the same nuclear background as HeLa cells, these results suggest that large-scale deletion mutations of mtDNA alone are sufficient for the mitochondrial dysfunction characteristic of CPEO.
在慢性进行性眼外肌麻痹(CPEO)患者中经常观察到具有大规模缺失的突变线粒体DNA(δ-mtDNA),CPEO是线粒体脑肌病的一个亚组。为了排除核基因组参与CPEO特征性线粒体功能障碍的表达,我们将一名CPEO患者的mtDNA导入无mtDNA的克隆HeLa细胞中,并分离出胞质杂种克隆。对胞质杂种中δ-mtDNA的定量分析表明,δ-mtDNA被选择性增殖,较高水平的δ-mtDNA与较慢的细胞生长速率相关。在这些胞质杂种克隆中,只有当δ-mtDNA占总mtDNA的比例小于60%时,缺失tRNA的翻译互补才会发生,而当δ-mtDNA积累到大于60%时,会导致整个线粒体群体的整体线粒体翻译逐渐受到抑制,以及细胞色素c氧化酶活性降低。由于这些胞质杂种与HeLa细胞具有相同的核背景,这些结果表明,仅mtDNA的大规模缺失突变就足以导致CPEO特征性的线粒体功能障碍。