Génétique moléculaire et défense antivirale, Centre National de la Recherche Scientifique, Université Paris Descartes, 45 rue des Saints Pères, 75006 Paris, France.
Brain Sci. 2014 Mar 3;4(1):91-122. doi: 10.3390/brainsci4010091.
Huntington disease and other diseases of polyglutamine expansion are each caused by a different protein bearing an excessively long polyglutamine sequence and are associated with neuronal death. Although these diseases affect largely different brain regions, they all share a number of characteristics, and, therefore, are likely to possess a common mechanism. In all of the diseases, the causative protein is proteolyzed, becomes abnormally folded and accumulates in oligomers and larger aggregates. The aggregated and possibly the monomeric expanded polyglutamine are likely to play a critical role in the pathogenesis and there is increasing evidence that the secondary structure of the protein influences its toxicity. We describe here, with special attention to huntingtin, the mechanisms of polyglutamine aggregation and the modulation of aggregation by the sequences flanking the polyglutamine. We give a comprehensive picture of the characteristics of monomeric and aggregated polyglutamine, including morphology, composition, seeding ability, secondary structure, and toxicity. The structural heterogeneity of aggregated polyglutamine may explain why polyglutamine-containing aggregates could paradoxically be either toxic or neuroprotective.
亨廷顿病和其他多聚谷氨酰胺扩展疾病分别由携带过长多聚谷氨酰胺序列的不同蛋白质引起,并与神经元死亡有关。尽管这些疾病主要影响不同的脑区,但它们都具有一些共同的特征,因此可能具有共同的机制。在所有这些疾病中,致病蛋白被蛋白水解酶切割,变得异常折叠,并在寡聚体和更大的聚集体中积累。聚集的、可能是单体的扩展多聚谷氨酰胺很可能在发病机制中发挥关键作用,越来越多的证据表明蛋白质的二级结构影响其毒性。我们在这里特别关注亨廷顿病,描述了多聚谷氨酰胺聚集的机制以及多聚谷氨酰胺侧翼序列对聚集的调节。我们全面描述了单体和聚集的多聚谷氨酰胺的特征,包括形态、组成、成核能力、二级结构和毒性。聚集的多聚谷氨酰胺的结构异质性可以解释为什么含有多聚谷氨酰胺的聚集体可以既是有毒的又是神经保护的。