Janer Alexandre, Martin Elodie, Muriel Marie-Paule, Latouche Morwena, Fujigasaki Hiroto, Ruberg Merle, Brice Alexis, Trottier Yvon, Sittler Annie
Institut National de la Santé et de la Recherche Médicale U679, Neurologie et Thérapeutique Expérimentale, 75651 Paris Cedex 13, France.
J Cell Biol. 2006 Jul 3;174(1):65-76. doi: 10.1083/jcb.200511045.
The pathogenesis of spinocerebellar ataxia type 7 and other neurodegenerative polyglutamine (polyQ) disorders correlates with the aberrant accumulation of toxic polyQ-expanded proteins in the nucleus. Promyelocytic leukemia protein (PML) nuclear bodies are often present in polyQ aggregates, but their relation to pathogenesis is unclear. We show that expression of PML isoform IV leads to the formation of distinct nuclear bodies enriched in components of the ubiquitin-proteasome system. These bodies recruit soluble mutant ataxin-7 and promote its degradation by proteasome-dependent proteolysis, thus preventing the aggregate formation. Inversely, disruption of the endogenous nuclear bodies with cadmium increases the nuclear accumulation and aggregation of mutant ataxin-7, demonstrating their role in ataxin-7 turnover. Interestingly, beta-interferon treatment, which induces the expression of endogenous PML IV, prevents the accumulation of transiently expressed mutant ataxin-7 without affecting the level of the endogenous wild-type protein. Therefore, clastosomes represent a potential therapeutic target for preventing polyQ disorders.
7型脊髓小脑共济失调及其他神经退行性多聚谷氨酰胺(polyQ)疾病的发病机制与有毒的多聚谷氨酰胺扩展蛋白在细胞核中的异常积累相关。早幼粒细胞白血病蛋白(PML)核体常存在于多聚谷氨酰胺聚集体中,但其与发病机制的关系尚不清楚。我们发现,PML异构体IV的表达会导致富含泛素-蛋白酶体系统成分的独特核体形成。这些核体招募可溶性突变型共济失调蛋白7,并通过蛋白酶体依赖性蛋白水解促进其降解,从而防止聚集体形成。相反,用镉破坏内源性核体会增加突变型共济失调蛋白7的核内积累和聚集,证明它们在共济失调蛋白7周转中的作用。有趣的是,诱导内源性PML IV表达的β-干扰素治疗可防止瞬时表达的突变型共济失调蛋白7积累,而不影响内源性野生型蛋白水平。因此,分裂体是预防多聚谷氨酰胺疾病的潜在治疗靶点。