Suppr超能文献

常染色体显性小脑共济失调。

Autosomal dominant cerebellar ataxias.

机构信息

Département de génétique et cytogénétique, consultation de génétique clinique, CHU Pitié-Salpêtrière, AP-HP, 47, boulevard de l'Hôpital, 75013 Paris, France.

出版信息

Rev Neurol (Paris). 2011 May;167(5):385-400. doi: 10.1016/j.neurol.2011.01.015. Epub 2011 May 5.

Abstract

Cerebellar ataxias with autosomal dominant transmission (ADCA) are far rarer than sporadic cases of cerebellar ataxia. The identification of genes involved in dominant forms has confirmed the genetic heterogeneity of these conditions and of the underlying mechanisms and pathways. To date, at least 28 genetic loci and, among them, 20 genes have been identified. In many instances, the phenotype is not restricted to cerebellar dysfunction but includes more complex multisystemic neurological deficits. Seven ADCA (SCA1, 2, 3, 6, 7, 17, and dentatorubro-pallido-luysian atrophy) are caused by repeat expansions in the corresponding proteins; phenotype-genotype correlations have shown that repeat size influences the progression of the disease, its severity and clinical differences among patients, including the phenomenon of anticipation between generations. All other ADCA are caused either by non-coding repeat expansions, conventional mutations or large rearrangements in genes with different functions. This review will focus on the genetic features of ADCA and on the clinical differences among the different forms.

摘要

常染色体显性遗传小脑共济失调(ADCA)远比散发性小脑共济失调少见。涉及显性形式的基因的鉴定证实了这些病症以及潜在机制和途径的遗传异质性。迄今为止,已经确定了至少 28 个遗传位点,其中包括 20 个基因。在许多情况下,表型不仅限于小脑功能障碍,还包括更复杂的多系统神经缺陷。七种 ADCA(SCA1、2、3、6、7、17 和齿状核红核苍白球路易体萎缩)是由相应蛋白中的重复扩展引起的;表型-基因型相关性表明,重复大小影响疾病的进展、其严重程度以及患者之间的临床差异,包括代际间的预期现象。所有其他 ADCA 要么是由非编码重复扩展、常规突变或具有不同功能的基因中的大片段重排引起的。这篇综述将重点介绍 ADCA 的遗传特征以及不同形式之间的临床差异。

文献AI研究员

20分钟写一篇综述,助力文献阅读效率提升50倍。

立即体验

用中文搜PubMed

大模型驱动的PubMed中文搜索引擎

马上搜索

文档翻译

学术文献翻译模型,支持多种主流文档格式。

立即体验