Brown P, Goldfarb L G, Gibbs C J, Gajdusek D C
Laboratory of CNS Studies, NINDS, NIH, Bethesda, Maryland 20892.
Eur J Epidemiol. 1991 Sep;7(5):469-76. doi: 10.1007/BF00143124.
Cases of familial Creutzfeldt-Jakob disease (CJD) with mutations in the PRNP gene were analyzed for distinctive clinico-pathological and experimental transmission characteristics. An insert mutation within the region of codons 51 to 91 was associated with a markedly early age at onset and prolonged course of illness. Point mutations at codons 178 and 200 were also associated with ages at onset, durations of illness, and clinical symptom profiles that differed from sporadic CJD. The age at onset of illness in each group was correlated with the length of incubation periods in primates inoculated with their brain tissue, suggesting that the early onset of familial CJD results not from a time shift of the initiating event, but from an accelerated pre-clinical (incubation) phase of disease, perhaps due to a more rapid formation of amyloid induced by a mutationally-altered precursor protein template.
对具有PRNP基因突变的家族性克雅氏病(CJD)病例进行了分析,以研究其独特的临床病理和实验性传播特征。密码子51至91区域内的插入突变与发病年龄显著提前和病程延长有关。密码子178和200处的点突变也与发病年龄、病程以及不同于散发性CJD的临床症状谱有关。每组疾病的发病年龄与接种其脑组织的灵长类动物的潜伏期长度相关,这表明家族性CJD的早发不是由于起始事件的时间转移,而是由于疾病临床前期(潜伏期)阶段加速,这可能是由于突变改变的前体蛋白模板诱导淀粉样蛋白形成更快所致。