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遗传性朊病毒病:美国一家快速进展性痴呆中心的经验及文献综述

Genetic prion disease: Experience of a rapidly progressive dementia center in the United States and a review of the literature.

作者信息

Takada Leonel T, Kim Mee-Ohk, Cleveland Ross W, Wong Katherine, Forner Sven A, Gala Ignacio Illán, Fong Jamie C, Geschwind Michael D

机构信息

Department of Neurology, Cognitive and Behavioral Neurology Unit, University of São Paulo, São Paulo, Brazil.

Department of Neurology, Memory and Aging Center, University of California, San Francisco, California.

出版信息

Am J Med Genet B Neuropsychiatr Genet. 2017 Jan;174(1):36-69. doi: 10.1002/ajmg.b.32505.

Abstract

Although prion diseases are generally thought to present as rapidly progressive dementias with survival of only a few months, the phenotypic spectrum for genetic prion diseases (gPrDs) is much broader. The majority have a rapid decline with short survival, but many patients with gPrDs present as slowly progressive ataxic or parkinsonian disorders with progression over a few to several years. A few very rare mutations even present as neuropsychiatric disorders, sometimes with systemic symptoms such as gastrointestinal disorders and neuropathy, progressing over years to decades. gPrDs are caused by mutations in the prion protein gene (PRNP), and have been historically classified based on their clinicopathological features as genetic Jakob-Creutzfeldt disease (gJCD), Gerstmann-Sträussler-Scheinker (GSS), or Fatal Familial Insomnia (FFI). Mutations in PRNP can be missense, nonsense, and octapeptide repeat insertions or a deletion, and present with diverse clinical features, sensitivities of ancillary testing, and neuropathological findings. We present the UCSF gPrD cohort, including 129 symptomatic patients referred to and/or seen at UCSF between 2001 and 2016, and compare the clinical features of the gPrDs from 22 mutations identified in our cohort with data from the literature, as well as perform a literature review on most other mutations not represented in our cohort. E200K is the most common mutation worldwide, is associated with gJCD, and was the most common in the UCSF cohort. Among the GSS-associated mutations, P102L is the most commonly reported and was also the most common at UCSF. We also had several octapeptide repeat insertions (OPRI), a rare nonsense mutation (Q160X), and three novel mutations (K194E, E200G, and A224V) in our UCSF cohort. © 2016 Wiley Periodicals, Inc.

摘要

尽管朊病毒疾病通常被认为表现为快速进展的痴呆症,患者生存期仅几个月,但遗传型朊病毒疾病(gPrDs)的表型谱要广泛得多。大多数患者病情迅速恶化,生存期短,但许多gPrDs患者表现为缓慢进展的共济失调或帕金森氏症,病程持续数年至数年。少数非常罕见的突变甚至表现为神经精神疾病,有时伴有胃肠道疾病和神经病变等全身症状,病程长达数年至数十年。gPrDs由朊病毒蛋白基因(PRNP)突变引起,历史上根据其临床病理特征分为遗传性克雅氏病(gJCD)、格斯特曼-施特劳斯勒-谢inker病(GSS)或致死性家族性失眠症(FFI)。PRNP突变可以是错义突变、无义突变、八肽重复插入或缺失,表现出不同的临床特征、辅助检查敏感性和神经病理学发现。我们展示了加州大学旧金山分校的gPrD队列,包括2001年至2016年间转诊至该校和/或在该校就诊的129例有症状患者,并将我们队列中鉴定出的22种突变的gPrDs临床特征与文献数据进行比较,同时对我们队列中未出现的大多数其他突变进行文献综述。E200K是全球最常见的突变,与gJCD相关,也是加州大学旧金山分校队列中最常见的突变。在与GSS相关的突变中,P102L是最常报道且在加州大学旧金山分校也是最常见的。我们的加州大学旧金山分校队列中还有几个八肽重复插入(OPRI)、一个罕见的无义突变(Q160X)和三个新突变(K194E、E200G和A224V)。© 2016威利期刊公司

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