Gómez-Durán Aurora, Pacheu-Grau David, Martínez-Romero Iñigo, López-Gallardo Ester, López-Pérez Manuel J, Montoya Julio, Ruiz-Pesini Eduardo
Departamento de Bioquímica, Biología Molecular y Celular, Centro de Investigaciones Biomédicas En Red de Enfermedades Raras, Universidad de Zaragoza, Spain.
Biochim Biophys Acta. 2012 Aug;1822(8):1216-22. doi: 10.1016/j.bbadis.2012.04.014. Epub 2012 Apr 26.
Leber's hereditary optic neuropathy is a maternally inherited optic atrophy caused by mitochondrial DNA point mutations. Previous epidemiological studies have shown that individuals from mitochondrial genetic backgrounds (haplogroups) J/Uk and H have a higher and a lower risk, respectively, of suffering this disorder. To analyze the bases of these associations at cellular and molecular levels, functional studies with cybrids provide high quality evidence. Cybrids from haplogroup J contain less mitochondrial deoxyribonucleic acid (DNA) and ribonucleic acid (RNA) and synthesize a smaller amount of mitochondrial DNA-encoded polypeptides than those from haplogroup H. Haplogroup J cybrids also display lower oxygen consumption, mitochondrial inner membrane potential and total adenosine-5'-triphosphate (ATP) levels. Moreover, mitochondrial DNA levels correlate with many parameters of the oxidative phosphorylation system. These results suggest that the mitochondrial DNA amount determines oxidative phosphorylation capacity and, along with other recently published observations, support the possibility that mitochondrial DNA levels may be responsible for the bias of the disorder toward males, for the incomplete penetrance of mutations causing Leber's hereditary optic neuropathy and for the association of the disease with particular mitochondrial DNA haplogroups.
莱伯遗传性视神经病变是一种由线粒体DNA点突变引起的母系遗传的视神经萎缩。先前的流行病学研究表明,来自线粒体遗传背景(单倍群)J/Uk和H的个体分别具有较高和较低的患该疾病的风险。为了在细胞和分子水平上分析这些关联的基础,利用胞质杂种进行的功能研究提供了高质量的证据。与来自单倍群H的胞质杂种相比,来自单倍群J的胞质杂种含有更少的线粒体脱氧核糖核酸(DNA)和核糖核酸(RNA),并且合成的线粒体DNA编码多肽量更少。单倍群J胞质杂种还表现出较低的耗氧量、线粒体内膜电位和总腺苷-5'-三磷酸(ATP)水平。此外,线粒体DNA水平与氧化磷酸化系统的许多参数相关。这些结果表明,线粒体DNA的量决定了氧化磷酸化能力,并且与最近发表的其他观察结果一起,支持了线粒体DNA水平可能导致该疾病在男性中更易发病、导致莱伯遗传性视神经病变的突变不完全外显以及该疾病与特定线粒体DNA单倍群相关联的可能性。