Goldfarb L G, Brown P, McCombie W R, Goldgaber D, Swergold G D, Wills P R, Cervenakova L, Baron H, Gibbs C J, Gajdusek D C
Laboratory of Central Nervous System Studies, National Institute of Neurological Disorders and Stroke, National Institutes of Health, Bethesda, MD 20892.
Proc Natl Acad Sci U S A. 1991 Dec 1;88(23):10926-30. doi: 10.1073/pnas.88.23.10926.
The PRNP gene, encoding the amyloid precursor protein that is centrally involved in Creutzfeldt-Jakob disease (CJD), has an unstable region of five variant tandem octapeptide coding repeats between codons 51 and 91. We screened a total of 535 individuals for the presence of extra repeats in this region, including patients with sporadic and familial forms of spongiform encephalopathy, members of their families, other neurological and non-neurological patients, and normal controls. We identified three CJD families (in each of which the proband's disease was neuropathologically confirmed and experimentally transmitted to primates) that were heterozygous for alleles with 10, 12, or 13 repeats, some of which had "wobble" nucleotide substitutions. We also found one individual with 9 repeats and no nucleotide substitutions who had no evidence of neurological disease. These observations, together with data on published British patients with 11 and 14 repeats, strongly suggest that the occurrence of 10 or more octapeptide repeats in the encoded amyloid precursor protein predisposes to CJD.
朊蛋白基因(PRNP)编码的淀粉样前体蛋白在克雅氏病(CJD)中起核心作用,该基因在密码子51至91之间有一个由五个可变串联八肽编码重复序列组成的不稳定区域。我们对总共535名个体进行了筛查,以确定该区域是否存在额外重复序列,这些个体包括散发性和家族性海绵状脑病患者、他们的家庭成员、其他神经科和非神经科患者以及正常对照。我们鉴定出三个CJD家族(每个家族中先证者的疾病均经神经病理学证实并通过实验传播给灵长类动物),这些家族对于具有10、12或13个重复序列的等位基因是杂合的,其中一些具有“摆动”核苷酸替换。我们还发现一名个体有9个重复序列且无核苷酸替换,没有神经疾病的证据。这些观察结果,连同已发表的具有11和14个重复序列的英国患者的数据,强烈表明编码的淀粉样前体蛋白中出现10个或更多八肽重复序列易患CJD。