Wong A, Cortopassi G
Department of Molecular Biosciences, University of California, Davis 95616, USA.
Biochem Biophys Res Commun. 1997 Oct 9;239(1):139-45. doi: 10.1006/bbrc.1997.7443.
The complete mechanism by which pathogenic mtDNA mutations cause cellular pathophysiology and in some cases cell death is unclear. Oxidant stress is especially toxic to excitable nerve and muscle cells, cells that are often affected in mitochondrial disease. The sensitivity of cells bearing the LHON, MELAS, and MERRF mutations to oxidant stress was determined. All were significantly more sensitive to H2O2 exposure than their nonmutant cybrid controls, the order of sensitivity was MELAS > LHON > MERRF > controls. Depletion of Ca2+ from the medium protected all cell lines from oxidant stress, consistent with the hypothesis that death induced by oxidant stress is Ca(2+)-dependent. A potential downstream target of Ca2+ is the mitochondrial permeability transition, MPT, which is inhibited by cyclosporin A. Treatment of MELAS, LHON, and MERRF cells with cyclosporin A caused significant rescue from oxidant exposure, and in each case significantly greater rescue of mutant than control cells. The pronounced oxidant-sensitivity of mutant cells, and their protection by Ca2+ depletion and CsA, has potential implications for both the pathophysiological mechanism and therapy of these mitochondrial genetic diseases.
致病性线粒体DNA(mtDNA)突变导致细胞病理生理变化,在某些情况下导致细胞死亡的完整机制尚不清楚。氧化应激对可兴奋的神经和肌肉细胞尤其有毒,而这些细胞常常在线粒体疾病中受到影响。研究人员测定了携带Leber遗传性视神经病变(LHON)、线粒体脑肌病伴乳酸血症和卒中样发作(MELAS)以及肌阵挛性癫痫伴破碎红纤维病(MERRF)突变的细胞对氧化应激的敏感性。所有这些细胞系对过氧化氢(H2O2)暴露的敏感性均显著高于其非突变胞质杂种对照,敏感性顺序为MELAS>LHON>MERRF>对照。从培养基中去除钙离子(Ca2+)可保护所有细胞系免受氧化应激,这与氧化应激诱导的细胞死亡是钙(Ca2+)依赖性的假说一致。Ca2+的一个潜在下游靶点是线粒体通透性转换(MPT),而环孢菌素A可抑制MPT。用环孢菌素A处理MELAS、LHON和MERRF细胞可显著减轻氧化应激暴露的影响,并且在每种情况下,突变细胞的恢复程度均显著高于对照细胞。突变细胞对氧化应激的显著敏感性以及钙离子去除和环孢菌素A对它们的保护作用,对于这些线粒体遗传病的病理生理机制和治疗均具有潜在意义。