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一个携带PRNP-P102L突变的新家族中具有可变表型的格斯特曼-施特劳斯勒-谢inker综合征。

Gerstmann-Sträussler-Scheinker syndrome with variable phenotype in a new kindred with PRNP-P102L mutation.

作者信息

Riudavets Miguel A, Sraka María Alejandra, Schultz Marcelo, Rojas Estefanía, Martinetto Horacio, Begué Christian, Noher de Halac Inés, Poleggi Anna, Equestre Michele, Pocchiari Maurizio, Sevlever Gustavo, Taratuto Ana Lía

机构信息

Department of Neuropathology, Institute for Neurological Research, FLENI, Buenos Aires, Argentina.

出版信息

Brain Pathol. 2014 Mar;24(2):142-7. doi: 10.1111/bpa.12083. Epub 2013 Sep 19.

Abstract

Gerstmann-Sträussler-Scheinker syndrome (GSS) is a dominantly inherited disorder belonging to the group of transmissible human spongiform encephalopathies or prion diseases. Several families affected by GSS with patients carrying mutations in the prion protein gene have been described worldwide. We report clinical, genealogical, neuropathology and molecular study results from two members of the first Argentine kindred affected by GSS. Both family members presented a frontotemporal-like syndrome, one with and the other without ataxia, with different lesions on neuropathology. A Pro to Leu point mutation at codon 102 (P102L) of the prion protein gene was detected in one of the subjects studied. The pathogenic basis of phenotypic variability observed in this family remains unclear, but resembles that observed in other P102L GSS patients from the same family.

摘要

格斯特曼-施特劳斯勒-谢inker综合征(GSS)是一种常染色体显性遗传性疾病,属于人类传染性海绵状脑病或朊病毒病。全球已报道了几个受GSS影响的家族,其患者的朊病毒蛋白基因发生了突变。我们报告了阿根廷首个受GSS影响的家族中两名成员的临床、系谱、神经病理学和分子研究结果。两名家族成员均表现出类似额颞叶的综合征,其中一名有共济失调,另一名没有,神经病理学上有不同的病变。在所研究的一名受试者中检测到朊病毒蛋白基因第102密码子(P102L)处的脯氨酸到亮氨酸点突变。该家族中观察到的表型变异性的致病基础尚不清楚,但与来自同一家族的其他P102L GSS患者中观察到的情况相似。

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