Medical Research Council (MRC) Prion Unit, University College London (UCL) Institute of Neurology, London, United Kingdom.
Proc Natl Acad Sci U S A. 2012 Aug 21;109(34):13722-7. doi: 10.1073/pnas.1208917109. Epub 2012 Aug 6.
Prion diseases are fatal neurodegenerative disorders that include bovine spongiform encephalopathy (BSE) and scrapie in animals and Creutzfeldt-Jakob disease (CJD) in humans. They are characterized by long incubation periods, variation in which is determined by many factors including genetic background. In some cases it is possible that incubation time may be directly correlated to the level of gene expression. To test this hypothesis, we combined incubation time data from five different inbred lines of mice with quantitative gene expression profiling in normal brains and identified five genes with expression levels that correlate with incubation time. One of these genes, Hspa13 (Stch), is a member of the Hsp70 family of ATPase heat shock proteins, which have been previously implicated in prion propagation. To test whether Hspa13 plays a causal role in determining the incubation period, we tested two overexpressing mouse models. The Tc1 human chromosome 21 (Hsa21) transchromosomic mouse model of Down syndrome is trisomic for many Hsa21 genes including Hspa13 and following Chandler/Rocky Mountain Laboratory (RML) prion inoculation, shows a 4% reduction in incubation time. Furthermore, a transgenic model with eightfold overexpression of mouse Hspa13 exhibited highly significant reductions in incubation time of 16, 15, and 7% following infection with Chandler/RML, ME7, and MRC2 prion strains, respectively. These data further implicate Hsp70-like molecular chaperones in protein misfolding disorders such as prion disease.
朊病毒病是致命的神经退行性疾病,包括动物中的牛海绵状脑病(BSE)和瘙痒病,以及人类中的克雅氏病(CJD)。它们的特征是潜伏期长,潜伏期的变化由许多因素决定,包括遗传背景。在某些情况下,潜伏期可能与基因表达水平直接相关。为了验证这一假设,我们将来自五个不同近交系小鼠的潜伏期数据与正常大脑的定量基因表达谱相结合,鉴定出与潜伏期相关的五个表达水平发生变化的基因。其中一个基因 Hspa13(Stch)是 Hsp70 家族 ATP 酶热休克蛋白的成员,先前已被牵连到朊病毒的传播中。为了验证 Hspa13 是否在确定潜伏期方面起因果作用,我们测试了两个过表达小鼠模型。唐氏综合征的 Tc1 人类 21 号染色体(Hsa21)转染色体小鼠模型在 Hsa21 基因上是三体的,包括 Hspa13,并且在接受 Chandler/Rocky Mountain Laboratory(RML)朊病毒接种后,潜伏期缩短了 4%。此外,一个 Hspa13 过表达 8 倍的转基因模型在感染 Chandler/RML、ME7 和 MRC2 朊病毒株后,潜伏期分别显著缩短了 16%、15%和 7%。这些数据进一步表明 Hsp70 样分子伴侣在朊病毒病等蛋白质错误折叠疾病中发挥作用。