Literature DB >> 29938355

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

Helena Fussiger1, Maria Luiza Saraiva-Pereira2,3,4, Sandra Leistner-Segal1,4, Laura Bannach Jardim5,6,7,8.   

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.

Entities:  

Keywords:  Diagnostic yield; FRDA; FXN; Friedreich ataxia; Prevalence

Mesh:

Substances:

Year:  2019        PMID: 29938355     DOI: 10.1007/s12311-018-0958-x

Source DB:  PubMed          Journal:  Cerebellum        ISSN: 1473-4222            Impact factor:   3.847


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Journal:  Can J Neurol Sci       Date:  1979-05       Impact factor: 2.104

Review 3.  Prevalence gradients of Friedreich's ataxia and R1b haplotype in Europe co-localize, suggesting a common Palaeolithic origin in the Franco-Cantabrian ice age refuge.

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4.  Friedreich's Ataxia (FRDA) is an extremely rare cause of autosomal recessive ataxia in Chinese Han population.

Authors:  Junsheng Zeng; Junling Wang; Sheng Zeng; Miao He; Xianfeng Zeng; Yao Zhou; Zhen Liu; Hong Jiang; Beisha Tang
Journal:  J Neurol Sci       Date:  2015-03-06       Impact factor: 3.181

5.  Epidemiological, clinical, paraclinical and molecular study of a cohort of 102 patients affected with autosomal recessive progressive cerebellar ataxia from Alsace, Eastern France: implications for clinical management.

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Journal:  Neurogenetics       Date:  2009-05-14       Impact factor: 2.660

6.  Friedreich's ataxia: a clinical and genetic study of 90 families with an analysis of early diagnostic criteria and intrafamilial clustering of clinical features.

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Journal:  Brain       Date:  1981-09       Impact factor: 13.501

7.  Genetic admixture of European FRDA genes is the cause of Friedreich ataxia in the Mexican population.

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8.  Prevalence of inherited ataxias in the province of Padua, Italy.

Authors:  M Zortea; M Armani; E Pastorello; G F Nunez; S Lombardi; S Tonello; M T Rigoni; L Zuliani; M L Mostacciuolo; C Gellera; S Di Donato; C P Trevisan
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9.  Revisiting the genetic ancestry of Brazilians using autosomal AIM-Indels.

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Journal:  PLoS One       Date:  2013-09-20       Impact factor: 3.240

10.  Friedreich's ataxia: autosomal recessive disease caused by an intronic GAA triplet repeat expansion.

Authors:  V Campuzano; L Montermini; M D Moltò; L Pianese; M Cossée; F Cavalcanti; E Monros; F Rodius; F Duclos; A Monticelli; F Zara; J Cañizares; H Koutnikova; S I Bidichandani; C Gellera; A Brice; P Trouillas; G De Michele; A Filla; R De Frutos; F Palau; P I Patel; S Di Donato; J L Mandel; S Cocozza; M Koenig; M Pandolfo
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  2 in total

1.  Frequency and Genetic Profile of Compound Heterozygous Friedreich's Ataxia Patients-the Brazilian Experience.

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Journal:  Cerebellum       Date:  2019-12       Impact factor: 3.847

2.  Genetic Analysis of Hereditary Ataxias in Peru Identifies SCA10 Families with Incomplete Penetrance.

Authors:  Mario Cornejo-Olivas; Miguel Inca-Martinez; Raphael Machado Castilhos; Gabriel Vasata Furtado; Eduardo Preusser Mattos; Giovana Bavia Bampi; Sandra Leistner-Segal; Victoria Marca; Pilar Mazzetti; Maria Luiza Saraiva-Pereira; Laura Bannach Jardim
Journal:  Cerebellum       Date:  2020-04       Impact factor: 3.847

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