Smith Karen M, Matson Samantha, Matson Wayne R, Cormier Kerry, Del Signore Steven J, Hagerty Sean W, Stack Edward C, Ryu Hoon, Ferrante Robert J
Geriatric Research Education and Clinical Center, Bedford VA Medical Center, Bedford 01730, and Neurology Department, Boston University School of Medicine, MA 02180, USA.
Biochim Biophys Acta. 2006 Jun;1762(6):616-26. doi: 10.1016/j.bbadis.2006.03.004. Epub 2006 Apr 17.
There is substantial evidence that a bioenergetic defect may play a role in the pathogenesis of Huntington's Disease (HD). A potential therapy for remediating defective energy metabolism is the mitochondrial cofactor, coenzyme Q10 (CoQ10). We have reported that CoQ10 is neuroprotective in the R6/2 transgenic mouse model of HD. Based upon the encouraging results of the CARE-HD trial and recent evidence that high-dose CoQ10 slows the progressive functional decline in Parkinson's disease, we performed a dose ranging study administering high levels of CoQ10 from two commercial sources in R6/2 mice to determine enhanced efficacy. High dose CoQ10 significantly extended survival in R6/2 mice, the degree of which was dose- and source-dependent. CoQ10 resulted in a marked improvement in motor performance and grip strength, with a reduction in weight loss, brain atrophy, and huntingtin inclusions in treated R6/2 mice. Brain levels of CoQ10 and CoQ9 were significantly lower in R6/2 mice, in comparison to wild type littermate control mice. Oral administration of CoQ10 elevated CoQ10 plasma levels and significantly increased brain levels of CoQ9, CoQ10, and ATP in R6/2 mice, while reducing 8-hydroxy-2-deoxyguanosine concentrations, a marker of oxidative damage. We demonstrate that high-dose administration of CoQ10 exerts a greater therapeutic benefit in a dose dependent manner in R6/2 mice than previously reported and suggest that clinical trials using high dose CoQ10 in HD patients are warranted.
有大量证据表明生物能量缺陷可能在亨廷顿舞蹈症(HD)的发病机制中起作用。一种用于修复能量代谢缺陷的潜在疗法是线粒体辅因子辅酶Q10(CoQ10)。我们已经报道CoQ10在HD的R6/2转基因小鼠模型中具有神经保护作用。基于CARE-HD试验的令人鼓舞的结果以及最近的证据表明高剂量CoQ10可减缓帕金森病的进行性功能衰退,我们进行了一项剂量范围研究,给R6/2小鼠施用来自两种商业来源的高水平CoQ10,以确定增强的疗效。高剂量CoQ10显著延长了R6/2小鼠的生存期,其程度呈剂量和来源依赖性。CoQ10使R6/2小鼠的运动性能和握力有显著改善,体重减轻、脑萎缩和亨廷顿蛋白包涵体减少。与野生型同窝对照小鼠相比,R6/2小鼠脑内CoQ10和CoQ9水平显著降低。口服CoQ10可提高R6/2小鼠的血浆CoQ10水平,并显著增加脑内CoQ9、CoQ10和ATP水平,同时降低氧化损伤标志物8-羟基-2-脱氧鸟苷的浓度。我们证明,高剂量施用CoQ10在R6/2小鼠中以剂量依赖方式发挥比先前报道更大的治疗益处,并建议在HD患者中使用高剂量CoQ10进行临床试验是有必要的。