Campbell T A, Palmer M S, Will R G, Gibb W R, Luthert P J, Collinge J
Prion Disease Group, Department of Biochemistry and Molecular Genetics, St. Mary's Hospital Medical School, London, UK.
Neurology. 1996 Mar;46(3):761-6. doi: 10.1212/wnl.46.3.761.
There are coding mutations in the prion protein gene in familial Creutzfeldt-Jakob disease (CJD), Gerstmann-Straussler-Scheinker disease, and other phenotypes that make up the inherited prion diseases. Insertional mutations consisting of two, five, six, seven, eight, and nine additional octapeptide repeat elements are seen in the inherited prion diseases and usually present as atypical dementias with considerable intrafamilial phenotypic variability. A four-octarepeat insertion was reported previously in an individual without neurodegenerative disease who died of hepatic cirrhosis. Here we report a novel four-octarepeat insertional mutation in a case with classical clinical, electroencephalographic and histopathologic features of CJD with the unusual finding of pronounced prion protein immunoreactivity of the molecular layer of the cerebellum.
在家族性克雅氏病(CJD)、格斯特曼-施特劳斯勒-谢inker病以及构成遗传性朊病毒病的其他表型中,朊病毒蛋白基因存在编码突变。遗传性朊病毒病中可见由两个、五个、六个、七个、八个和九个额外八肽重复元件组成的插入突变,通常表现为具有显著家族内表型变异性的非典型痴呆。先前报道过一名死于肝硬化且无神经退行性疾病的个体存在四肽重复插入。在此,我们报告一例具有CJD典型临床、脑电图和组织病理学特征的病例中出现一种新的四肽重复插入突变,该病例有一个不寻常的发现,即小脑分子层有明显的朊病毒蛋白免疫反应性。