亚马孙地区(巴西北部)脊髓小脑共济失调病例:从地理、历史和遗传进化角度分析疾病频率。

IF 2.4 3区 医学 Q3 NEUROSCIENCES
Diana Vieira Brito, Marcus Vinicius Della Coletta, Giselle Benevides Monteiro Ferreira, Sabrina Rodrigues da Silva, Patricia Batista de Azevedo, Cleiton Fantin
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引用次数: 0

摘要

脊髓小脑共济失调(SCAs)是一组遗传性神经退行性疾病,在不同的种族和地理区域表现出不同的分布。巴西是一个人口高度混杂的大国,对SCAs流行情况的调查主要集中在有限的地区。在这里,我们描述了亚马逊州SCA类型的频率,以及SCA家族的地理起源,并将它们与巴西其他地区SCA频率的现有文献数据进行了比较。患者在玛瑙斯/亚马逊州的两个共济失调转诊中心招募。还根据当地医疗记录和来自其他机构的联系人积极搜索患者。对参与者进行ATXN1、ATXN2、ATXN3、CACNA1A、ATXN7和ATXN10基因扩增检测。总体而言,该人群中SCA阳性病例数较低(N = 18)。此外,SCA家族中最常见的类型是SCA2(30%),其次是SCA3(15%)、SCA7(15%)和SCA10(1例)。这些结果与巴西其他人群的结果形成对比,在巴西其他人群中,SCA3型占主导地位,占50%以上的病例。我们得出结论,亚马逊地区的地理隔离和独特的种群历史,包括强大的美洲印第安人血统和最近的人口增长,导致SCA具有多突变起源(SCA2)的频率更高,而不是通过祖先突变的欧洲迁移传播的SCA (SCA3)。我们认为,考虑这些因素是至关重要的,以更好地了解遗传疾病的流行病学。
本文章由计算机程序翻译,如有差异,请以英文原文为准。
The Case of Spinocerebellar Ataxias in Amazonas (Northern Brazil): An Analysis of Disease Frequency from a Geographic, Historical, and Genetic-Evolutionary Perspective.

The Spinocerebellar Ataxias (SCAs) are a group of hereditary neurodegenerative diseases that show a variable distribution among distinct ethnicities and geographic regions. In Brazil, a large and highly admixed country, the prevalence of SCAs has been investigated mostly in limited areas. Here we characterized the frequencies of SCA types in the state of Amazonas, as well as the geographic origin of SCA families, and compared them to the literature data available about the frequency of SCAs in other Brazilian regions. Patients were recruited at two referral centers for ataxias at Manaus/Amazonas. An active search of patients based on the local medical records and contacts from other institutions was also performed. The participants were genetically tested for expansions at ATXN1, ATXN2, ATXN3, CACNA1A, ATXN7 and ATXN10. Overall, the number of positive SCA cases was low in this population (N = 18). Also, the most frequent type among SCA families was SCA2 (30%), followed by SCA3 (15%), SCA7 (15%) and SCA10 (one case). These results contrast with those of other Brazilian populations, where SCA3 is predominant over the other types, comprising more than 50% of cases. We concluded that the geographic isolation and the unique population history of Amazonas, including strong Amerindian ancestry and recent demographic growth, have led to a higher frequency of an SCA with multiple mutation origins (SCA2) instead of a SCA spread by European migration of ancestral mutations (SCA3). We suggest that considering these factors is crucial for a better understanding of the epidemiology of hereditary diseases.

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来源期刊
Cerebellum
Cerebellum 医学-神经科学
CiteScore
6.40
自引率
14.30%
发文量
150
审稿时长
4-8 weeks
期刊介绍: Official publication of the Society for Research on the Cerebellum devoted to genetics of cerebellar ataxias, role of cerebellum in motor control and cognitive function, and amid an ageing population, diseases associated with cerebellar dysfunction. The Cerebellum is a central source for the latest developments in fundamental neurosciences including molecular and cellular biology; behavioural neurosciences and neurochemistry; genetics; fundamental and clinical neurophysiology; neurology and neuropathology; cognition and neuroimaging. The Cerebellum benefits neuroscientists in molecular and cellular biology; neurophysiologists; researchers in neurotransmission; neurologists; radiologists; paediatricians; neuropsychologists; students of neurology and psychiatry and others.
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