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弗里德赖希共济失调:巴西南部的诊断率和最小频率。

Friedreich Ataxia: Diagnostic Yield and Minimal Frequency in South Brazil.

机构信息

Programa de Pós-Graduação em Saúde da Criança e do Adolescente, Universidade Federal do Rio Grande do Sul, Rua Ramiro Barcelos, 2400, sala 220, Porto Alegre, 90035-003, Brazil.

Departamento de Bioquímica, Universidade Federal do Rio Grande do Sul, Rua Ramiro Barcelos, 2600-Prédio Anexo, Porto Alegre, 90035-003, Brazil.

出版信息

Cerebellum. 2019 Feb;18(1):147-151. doi: 10.1007/s12311-018-0958-x.

Abstract

Friedreich ataxia (FRDA) is an autosomal recessive disorder due to mutations in the FXN gene. FRDA is characterized by the classical triad of ataxia, absent reflexes, and Babinski sign, but atypical presentations might also occur. Our aims were to describe the proportion of FRDA diagnoses in suspected families living in Rio Grande do Sul, South Brazil, and to estimate a minimum frequency of symptomatic subjects. Subjects that were evaluated by molecular analysis for FRDA at the Hospital de Clínicas de Porto Alegre were identified in our files. Patients' clinical manifestation and phenotypes were described and compared. The number of FRDA subjects alive in the last 5 years was determined. One hundred fifty-six index cases (families) were submitted to evaluation of GAA repeats at FXN since 1997: 27 were confirmed as FRDA patients. Therefore, the diagnostic yield was 17.3%. Proportion of classical, late onset, and retained reflexes subphenotypes were similar to those described by other studies. A minimum prevalence was estimated as 0.20:100.000 inhabitants. In conclusion, we verified that this FRDA population displayed the usual clinical characteristics, but with a lower period prevalence than those obtained in populations from Europe.

摘要

弗里德赖希共济失调(FRDA)是一种常染色体隐性疾病,由 FXN 基因突变引起。FRDA 的特征为三联征:共济失调、反射消失和巴宾斯基征阳性,但也可能出现非典型表现。我们的目的是描述在巴西南里奥格兰德州疑似家族中 FRDA 诊断的比例,并估计有症状患者的最低频率。我们在文件中确定了在阿雷格里港临床医院因 FRDA 接受分子分析评估的患者。描述并比较了患者的临床表现和表型。确定了过去 5 年内存活的 FRDA 患者人数。自 1997 年以来,156 名索引病例(家族)接受了 FXN 中 GAA 重复的评估:27 名被确认为 FRDA 患者。因此,诊断率为 17.3%。经典、晚发和保留反射亚型的比例与其他研究描述的相似。估计最小患病率为 0.20:100000 居民。总之,我们发现该 FRDA 人群表现出通常的临床特征,但与欧洲人群获得的患病率相比要低。

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