Department of Biochemistry and Molecular Genetics, University of Virginia School of Medicine, Charlottesville, VA 22908, USA.
Proc Natl Acad Sci U S A. 2012 Dec 26;109(52):21319-24. doi: 10.1073/pnas.1218331110. Epub 2012 Dec 10.
Spinocerebellar ataxia type 7 (SCA7) is an autosomal-dominant neurodegenerative disorder that results from polyglutamine expansion of the ataxin-7 (ATXN7) protein. Remarkably, although mutant ATXN7 is expressed throughout the body, pathology is restricted primarily to the cerebellum and retina. One major goal has been to identify factors that contribute to the tissue specificity of SCA7. Here we describe the development and use of a human astrocyte cell culture model to identify reelin, a factor intimately involved in the development and maintenance of Purkinje cells and the cerebellum as a whole, as an ATXN7 target gene. We found that polyglutamine expansion decreased ATXN7 occupancy, which correlated with increased levels of histone H2B monoubiquitination, at the reelin promoter. Treatment with trichostatin A, but not other histone deacetylase inhibitors, partially restored reelin transcription and promoted the accumulation of mutant ATXN7 into nuclear inclusions. Our findings suggest that reelin could be a previously unknown factor involved in the tissue specificity of SCA7 and that trichostatin A may ameliorate deleterious effects of the mutant ATXN7 protein by promoting its sequestration away from promoters into nuclear inclusions.
脊髓小脑共济失调 7 型(SCA7)是一种常染色体显性遗传性神经退行性疾病,由ataxin-7(ATXN7)蛋白的多聚谷氨酰胺扩展引起。值得注意的是,尽管突变型 ATXN7 在全身表达,但病变主要局限于小脑和视网膜。一个主要目标是确定导致 SCA7 组织特异性的因素。在这里,我们描述了人类星形胶质细胞培养模型的开发和使用,以鉴定 reelin 作为 ATXN7 靶基因,reelin 是一种与 Purkinje 细胞的发育和维持以及整个小脑密切相关的因子。我们发现,多聚谷氨酰胺扩展降低了 reelin 启动子上 ATXN7 的占有率,这与组蛋白 H2B 单泛素化水平的增加相关。用曲古抑菌素 A 处理,而不是其他组蛋白去乙酰化酶抑制剂,部分恢复了 reelin 的转录,并促进了突变型 ATXN7 积累到核内包涵体中。我们的研究结果表明,reelin 可能是 SCA7 组织特异性的一个以前未知的因素,而曲古抑菌素 A 可能通过促进其从启动子隔离到核内包涵体中来减轻突变型 ATXN7 蛋白的有害影响。