Rosenmann H, Vardi J, Finkelstein Y, Chapman J, Gabizon R
Department of Neurology, Hadassah University Hospital, Jerusalem, Israel.
Acta Neurol Scand. 1998 Mar;97(3):184-7. doi: 10.1111/j.1600-0404.1998.tb00634.x.
Among the dozen known mutations in the PrP gene which segregate with the inherited prion diseases, only 2 mutations have been described in Israel so far: the codon 200 mutation in Creutzfeldt-Jakob disease (CJD) affected Libyan Jews, and the codon 102 mutation in 1 Jewish Gerstmann-Straussler-Scheinker (GSS) affected pedigree of German origin. We report here 2 unrelated CJD178 cases affected by a unique phenotype: aphemia, apraxia, uncontrolled laugh and no ataxia. As opposed to other CJD178 patients, in these patients, the signal transduction protein 14-3-3, recently suggested as a CJD marker, was detected in the cerebrospinal fluid samples by immunostaining. The D178N mutation, known to be linked to 2 different phenotypes: Fatal Familial Insomnia (FFI) and CJD, was not described so far among Jews. The phenotype reported here, although it shares a common Va1129/Asn178 haplotype with the previously described CJD178, may point to a different clinical subtype of CJD178.
在与遗传性朊病毒病相关的朊蛋白(PrP)基因已知的十几种突变中,到目前为止以色列仅描述了2种突变:在源自利比亚的克雅氏病(CJD)患者中发现的密码子200突变,以及在1个源自德国的犹太格斯特曼-施特劳斯勒-谢克尔综合征(GSS)家系中发现的密码子102突变。我们在此报告2例不相关的CJD178病例,其具有独特的表型:失音症、失用症、无法控制的大笑且无共济失调。与其他CJD178患者不同,在这些患者的脑脊液样本中,通过免疫染色检测到了最近被认为是CJD标志物的信号转导蛋白14-3-3。已知与2种不同表型相关的D178N突变:致死性家族性失眠症(FFI)和CJD,在犹太人中尚未有描述。这里报告的表型,尽管它与先前描述的CJD178共享常见的Va1129/Asn178单倍型,但可能指向CJD178的一种不同临床亚型。