Ehrnhoefer Dagmar E, Duennwald Martin, Markovic Phoebe, Wacker Jennifer L, Engemann Sabine, Roark Margaret, Legleiter Justin, Marsh J Lawrence, Thompson Leslie M, Lindquist Susan, Muchowski Paul J, Wanker Erich E
Max Delbrueck Center for Molecular Medicine, Department of Neuroproteomics, Berlin, Germany.
Hum Mol Genet. 2006 Sep 15;15(18):2743-51. doi: 10.1093/hmg/ddl210. Epub 2006 Aug 7.
Huntington's disease (HD) is a progressive neurodegenerative disorder for which only symptomatic treatments of limited effectiveness are available. Preventing early misfolding steps and thereby aggregation of the polyglutamine (polyQ)-containing protein huntingtin (htt) in neurons of patients may represent an attractive therapeutic strategy to postpone the onset and progression of HD. Here, we demonstrate that the green tea polyphenol (-)-epigallocatechin-3-gallate (EGCG) potently inhibits the aggregation of mutant htt exon 1 protein in a dose-dependent manner. Dot-blot assays and atomic force microscopy studies revealed that EGCG modulates misfolding and oligomerization of mutant htt exon 1 protein in vitro, indicating that it interferes with very early events in the aggregation process. Also, EGCG significantly reduced polyQ-mediated htt protein aggregation and cytotoxicity in an yeast model of HD. When EGCG was fed to transgenic HD flies overexpressing a pathogenic htt exon 1 protein, photoreceptor degeneration and motor function improved. These results indicate that modulators of htt exon 1 misfolding and oligomerization like EGCG are likely to reduce polyQ-mediated toxicity in vivo. Our studies may provide the basis for the development of a novel pharmacotherapy for HD and related polyQ disorders.
亨廷顿舞蹈症(HD)是一种进行性神经退行性疾病,目前仅有效果有限的对症治疗方法。防止早期错误折叠步骤,从而防止患者神经元中含多聚谷氨酰胺(polyQ)的亨廷顿蛋白(htt)聚集,可能是一种延缓HD发病和进展的有吸引力的治疗策略。在此,我们证明绿茶多酚(-)-表没食子儿茶素-3-没食子酸酯(EGCG)以剂量依赖的方式有效抑制突变型htt外显子1蛋白的聚集。斑点印迹分析和原子力显微镜研究表明,EGCG在体外调节突变型htt外显子1蛋白的错误折叠和寡聚化,表明它干扰聚集过程中的早期事件。此外,在HD酵母模型中,EGCG显著降低了polyQ介导的htt蛋白聚集和细胞毒性。当给过表达致病性htt外显子1蛋白的转基因HD果蝇喂食EGCG时,光感受器退化和运动功能得到改善。这些结果表明,像EGCG这样的htt外显子1错误折叠和寡聚化调节剂可能会降低体内polyQ介导的毒性。我们的研究可能为开发针对HD和相关polyQ疾病的新型药物疗法提供基础。