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伴有P102L致病突变的格斯特曼-施特劳斯勒-申克综合征表现为家族性克雅氏病:一例报告及文献复习

Gerstmann-Sträussler-Scheinker syndrome with the P102L pathogenic mutation presenting as familial Creutzfeldt-Jakob disease: a case report and review of the literature.

作者信息

Rusina Robert, Fiala Jindřich, Holada Karel, Matějčková Milada, Nováková Jana, Ampapa Radek, Koukolík František, Matěj Radoslav

机构信息

Department of Neurology, Thomayer Teaching Hospital and Institute for Postgraduate Education in Medicine, Prague, Czech Republic.

出版信息

Neurocase. 2013;19(1):41-53. doi: 10.1080/13554794.2011.654215. Epub 2012 Apr 12.

DOI:10.1080/13554794.2011.654215
PMID:22494260
Abstract

Gerstmann-Sträussler-Scheinker syndrome is a rare autosomal dominant disease caused by a mutation in the prion gene, usually manifesting as progressive ataxia with late cognitive decline. A 44-year-old woman with a positive family history developed early personality and behavior changes, followed by paresthesias and ataxia, later associated with memory problems, pyramidal signs, anosognosia and very late myoclonus, spasticity, and severe dysexecutive impairment. Magnetic resonance showed caudate, mesio-frontal, and insular hyper-intensities, electroencephalography revealed generalized triphasic periodic complexes. A pathogenic P102L mutation in the prion gene was detected. Our case differed from classical Gerstmann-Sträussler-Scheinker syndrome by rapid progression, severe dementia, abnormal electroencephalography and magnetic resonance findings, which were highly suggestive of familial Creutzfeldt-Jakob disease.

摘要

格斯特曼-施特劳斯勒-谢inker综合征是一种罕见的常染色体显性疾病,由朊病毒基因突变引起,通常表现为进行性共济失调并伴有晚期认知功能减退。一名有家族史阳性的44岁女性出现早期人格和行为改变,随后出现感觉异常和共济失调,后来伴有记忆问题、锥体束征、疾病感缺失以及非常晚期的肌阵挛、痉挛和严重的执行功能障碍。磁共振成像显示尾状核、内侧额叶和岛叶高信号,脑电图显示广泛性三相周期性复合波。检测到朊病毒基因中的致病性P102L突变。我们的病例与经典的格斯特曼-施特劳斯勒-谢inker综合征不同,其具有快速进展、严重痴呆、异常脑电图和磁共振成像表现,高度提示为家族性克雅氏病。

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Front Neurol. 2023 Aug 3;14:1187813. doi: 10.3389/fneur.2023.1187813. eCollection 2023.
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