Pan Tao, Li Ruliang, Kang Shin-Chung, Pastore Manuela, Wong Boon-Seng, Ironside James, Gambetti Pierluigi, Sy Man-Sun
Division of Neuropathology, Case Western Reserve University, Cleveland, Ohio 44120, USA.
J Neurochem. 2005 Jan;92(1):132-42. doi: 10.1111/j.1471-4159.2004.02859.x.
The phenotype of human prion diseases is influenced by the prion protein (PrP) genotype as determined by the methionine (M)/valine (V) polymorphism at codon 129, the scrapie PrP (PrPSc) type and the etiology. To gain further insight into the mechanisms of phenotype determination, we compared two-dimensional immunoblot profiles of detergent insoluble and proteinase K-resistant PrP species in a type of sporadic Creutzfeldt-Jakob disease (sCJDMM2), variant CJD (vCJD) and sporadic fatal insomnia (sFI). Full-length and truncated PrP forms present in the insoluble fractions were also separately analyzed. These three diseases were selected because they have the same M/M PrP genotype at codon 129 and the same type 2 PrPSc, but different etiologies, also sCJDMM2 and sFI are sporadic, whereas vCJD is acquired by infection. We observed minor differences in the PrP detergent-insoluble fractions between sCJDMM2 and vCJD, although both differ in the corresponding fractions from sFI. We detected more substantial heterogeneity between sCJDMM2 and vCJD in the two-dimensional blots of the proteinase K-resistant PrP fraction suggesting that different PrP species are selected for conversion to proteinase K-resistant PrP in sCJDMM2 and vCJD. These differences are mostly, but not exclusively, due to variations in the type of the N-linked glycans. We also show that the over-representation of the highly glycosylated forms distinctive of the proteinase K-resistant PrPSc of vCJD in one-dimensional blots is due to differences in both the amount and the natures of the glycans. Overall, these findings underline the complexity of phenotypic determination in human prion diseases.
人类朊病毒疾病的表型受朊病毒蛋白(PrP)基因型的影响,该基因型由密码子129处的甲硫氨酸(M)/缬氨酸(V)多态性、瘙痒病PrP(PrPSc)类型以及病因决定。为了进一步深入了解表型决定机制,我们比较了散发性克雅氏病(sCJDMM2)、变异型克雅氏病(vCJD)和散发性致死性失眠症(sFI)中去污剂不溶性和蛋白酶K抗性PrP物种的二维免疫印迹图谱。还分别分析了不溶性组分中存在的全长和截短的PrP形式。选择这三种疾病是因为它们在密码子129处具有相同的M/M PrP基因型和相同的2型PrPSc,但病因不同,此外,sCJDMM2和sFI是散发性的,而vCJD是通过感染获得的。我们观察到sCJDMM2和vCJD之间PrP去污剂不溶性组分存在细微差异,尽管两者在相应组分上与sFI不同。我们在蛋白酶K抗性PrP组分的二维印迹中检测到sCJDMM2和vCJD之间存在更显著的异质性,这表明在sCJDMM2和vCJD中,不同的PrP物种被选择转化为蛋白酶K抗性PrP。这些差异主要但并非完全是由于N-连接聚糖类型的变化。我们还表明,在一维印迹中vCJD的蛋白酶K抗性PrPSc特有的高糖基化形式的过度表达是由于聚糖的数量和性质的差异。总体而言,这些发现强调了人类朊病毒疾病表型决定的复杂性。