Hoffner Guylaine, Djian Philippe
Laboratoire de Physiologie Cérébrale, Centre National de la Recherche Scientifique, Université Paris Descartes, 45 rue des Saints Pères, 75006, Paris, France.
Mol Neurobiol. 2015 Dec;52(3):1297-1314. doi: 10.1007/s12035-014-8932-1. Epub 2014 Oct 22.
Huntington disease is a dominantly inherited disease of the central nervous system. The mutational expansion of polyglutamine beyond a critical length produces a toxic gain of function in huntingtin and results in neuronal death. In the course of the disease, expanded huntingtin is proteolyzed, becomes abnormally folded, and accumulates in oligomers, fibrils, and microscopic inclusions. The aggregated forms of the expanded protein are structurally diverse. Structural heterogeneity may explain why polyglutamine-containing aggregates could paradoxically be either toxic or neuroprotective. When defined, the toxic structures could then specifically be targeted by prophylactic or therapeutic drugs aimed at inhibiting polyglutamine aggregation.
亨廷顿舞蹈症是一种中枢神经系统的显性遗传病。聚谷氨酰胺的突变性扩增超过临界长度会在亨廷顿蛋白中产生毒性功能获得,并导致神经元死亡。在疾病过程中,扩增的亨廷顿蛋白被蛋白水解,发生异常折叠,并在寡聚体、原纤维和显微镜下可见的包涵体中积累。扩增蛋白的聚集形式在结构上多种多样。结构异质性或许可以解释为何含聚谷氨酰胺的聚集体会自相矛盾地既具有毒性又具有神经保护作用。一旦确定了毒性结构,那么旨在抑制聚谷氨酰胺聚集的预防性或治疗性药物就可以特异性地靶向这些结构。