Murayama Yuichi, Masujin Kentaro, Imamura Morikazu, Ono Fumiko, Shibata Hiroaki, Tobiume Minoru, Yamamura Tomoaki, Shimozaki Noriko, Terao Keiji, Yamakawa Yoshio, Sata Tetsutaro
Influenza and Prion Disease Research Center, National Institute of Animal Health, Tsukuba, Ibaraki, Japan.
Chiba Institute of Science Faculty of Risk and Crisis Management, Choshi, Chiba, Japan.
J Gen Virol. 2014 Nov;95(Pt 11):2576-2588. doi: 10.1099/vir.0.066225-0. Epub 2014 Jul 14.
Prion diseases are characterized by the prominent accumulation of the misfolded form of a normal cellular protein (PrP(Sc)) in the central nervous system. The pathological features and biochemical properties of PrP(Sc) in macaque monkeys infected with the bovine spongiform encephalopathy (BSE) prion have been found to be similar to those of human subjects with variant Creutzfeldt-Jakob disease (vCJD). Non-human primate models are thus ideally suited for performing valid diagnostic tests and determining the efficacy of potential therapeutic agents. In the current study, we developed a highly efficient method for in vitro amplification of cynomolgus macaque BSE PrP(Sc). This method involves amplifying PrP(Sc) by protein misfolding cyclic amplification (PMCA) using mouse brain homogenate as a PrP(C) substrate in the presence of sulfated dextran compounds. This method is capable of amplifying very small amounts of PrP(Sc) contained in the cerebrospinal fluid (CSF) and white blood cells (WBCs), as well as in the peripheral tissues of macaques that have been intracerebrally inoculated with the BSE prion. After clinical signs of the disease appeared in three macaques, we detected PrP(Sc) in the CSF by serial PMCA, and the CSF levels of PrP(Sc) tended to increase with disease progression. In addition, PrP(Sc) was detectable in WBCs at the clinical phases of the disease in two of the three macaques. Thus, our highly sensitive, novel method may be useful for furthering the understanding of the tissue distribution of PrP(Sc) in non-human primate models of CJD.
朊病毒疾病的特征是正常细胞蛋白的错误折叠形式(PrP(Sc))在中枢神经系统中显著积累。已发现感染牛海绵状脑病(BSE)朊病毒的猕猴体内PrP(Sc)的病理特征和生化特性与变异型克雅氏病(vCJD)患者的相似。因此,非人类灵长类动物模型非常适合进行有效的诊断测试并确定潜在治疗药物的疗效。在本研究中,我们开发了一种高效的食蟹猴BSE PrP(Sc)体外扩增方法。该方法包括在硫酸葡聚糖化合物存在的情况下,使用小鼠脑匀浆作为PrP(C)底物,通过蛋白质错误折叠循环扩增(PMCA)来扩增PrP(Sc)。这种方法能够扩增脑脊液(CSF)和白细胞(WBC)中以及经脑内接种BSE朊病毒的猕猴外周组织中所含的极少量PrP(Sc)。在三只猕猴出现疾病临床症状后,我们通过连续PMCA在脑脊液中检测到了PrP(Sc),并且脑脊液中PrP(Sc)的水平倾向于随着疾病进展而升高。此外,在三只猕猴中的两只处于疾病临床阶段的白细胞中也可检测到PrP(Sc)。因此,我们这种高度灵敏的新方法可能有助于进一步了解PrP(Sc)在非人类灵长类动物克雅氏病模型中的组织分布。