Bratosiewicz-Wasik Jolanta, Liberski Pawel P, Golanska Ewa, Jansen Gerhard H, Wasik Tomasz J
Department of Virology, Medical University of Silesia, 41-200 Sosnowiec, Narcyzow 1, Poland, and Department of Pathology and Laboratory Medicine, The Ottawa Hospital, Canada.
Neurosci Lett. 2007 Jan 16;411(3):163-7. doi: 10.1016/j.neulet.2006.08.001. Epub 2006 Nov 28.
The prion diseases are fatal neurodegenerative disorders that afflict both humans and animals. They comprise kuru, Creutzfeldt-Jakob disease (CJD), Gerstmman-Straussler-Scheinker syndrome (GSS), and fatal familial insomnia (FFI). Both GSS, FFI and approximately 10% of CJD cases are genetically linked disorders, whereas 90% of CJD cases are not associated with mutations in the PRNP coding region, therefore other factors must be involved in pathogenesis of these forms of CJD. There is strong evidence that in transgenic mice the level of PrP gene expression influences the initiation and progression of the prion diseases. Moreover, in in vitro experiments demonstrated that mutations in the regulatory region of PRNP gene altered gene expression, therefore it may be expected that PrP expression level influences the susceptibility to CJD. In order to investigate whether single nucleotide polymorphisms within regulatory region of PRNP may modulate genetic susceptibility to sporadic CJD we examined an association of the C/G polymorphism at position -101 with the sCJD. In our study -101G polymorphism is over-represented among sCJD PRNP codon 129M/V cases compared with the control group. Our data suggest that polymorphism at position -101 in the regulatory region of PRNP may be a risk factor for sCJD among codon 129 heterozygotes.
朊病毒疾病是一种致命的神经退行性疾病,可侵袭人类和动物。它们包括库鲁病、克雅氏病(CJD)、格斯特曼-施特劳斯勒-谢inker综合征(GSS)和致死性家族性失眠症(FFI)。GSS、FFI以及约10%的CJD病例都是与遗传相关的疾病,而90%的CJD病例与PRNP编码区的突变无关,因此这些形式的CJD发病机制中必定涉及其他因素。有强有力的证据表明,在转基因小鼠中,PrP基因表达水平会影响朊病毒疾病的起始和进展。此外,体外实验表明PRNP基因调控区的突变会改变基因表达,因此可以预期PrP表达水平会影响对CJD的易感性。为了研究PRNP调控区内的单核苷酸多态性是否可能调节散发性CJD的遗传易感性,我们检测了-101位C/G多态性与散发性CJD的关联。在我们的研究中,与对照组相比,-101G多态性在散发性CJD PRNP密码子129M/V病例中过度表达。我们的数据表明,PRNP调控区-101位的多态性可能是密码子129杂合子中散发性CJD的一个危险因素。