Literature DB >> 30232781

Description of rare mutations and a novel variant in Brazilian patients with Cystic Fibrosis: a case series from a referral center in the Bahia State.

Laís Ribeiro Mota1, Valmir Machado de Melo Filho2, Lorena Lemos de Castro2, Daniel Fantozzi Garcia3, Regina Terse-Ramos4, Maria Betânia Pereira Toralles4, Renata Lúcia Leite Ferreira de Lima5, Edna Lúcia Souza6.   

Abstract

Knowledge of the genetic profile of Cystic Fibrosis (CF) contributes to a better understanding of the genotype/phenotype relationship, particularly in mixed populations such as in Brazil. To describe clinical data of CF patients with rare or not yet observed CFTR gene mutations in Brazil. It was a case series of CF patients followed-up at a referral center. Clinical and laboratory data were obtained through medical records. Molecular analysis of the mutations was performed by conventional methods and/or by next-generation sequencing. Ten patients were studied, seven had five pathogenic mutations without previous description in Brazil (Q1100P, Y109C, A107P, E1409K and K162E), one of which has not yet been reported in patients with CF (A107P). Among the seven patients, three (two siblings) had the second mutant allele of rare occurrence among Brazilians patients (G1069R and 2307insA). Three other patients also had at least one rare variant (V201M, S466X and G1069R). The age of the CF diagnosis ranged from 1 to 190 months in the ten cases and the main clinical manifestations were respiratory symptoms and difficulty in gaining weight. All but one patient presented clinical and/or laboratory data compatible with pancreatic insufficiency. The identification of rare or not yet described CFTR mutations in patients with CF in Brazil highlights the high genetic heterogeneity in this population. Knowledge of the genotypic profile of Brazilian CF patients can contribute to the development of specific mutation panels for the genetic investigation targeting each region of the country, as well as helping to understand the complex genotype/phenotype relationship, especially in mixed populations.

Entities:  

Keywords:  CFTR; Genotype; Mucoviscidosis; Mutation

Mesh:

Substances:

Year:  2018        PMID: 30232781     DOI: 10.1007/s11033-018-4361-y

Source DB:  PubMed          Journal:  Mol Biol Rep        ISSN: 0301-4851            Impact factor:   2.316


  15 in total

Review 1.  Progress in therapies for cystic fibrosis.

Authors:  Kris De Boeck; Margarida D Amaral
Journal:  Lancet Respir Med       Date:  2016-04-01       Impact factor: 30.700

2.  Analysis of the CFTR gene confirms the high genetic heterogeneity of the Spanish population: 43 mutations account for only 78% of CF chromosomes.

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3.  Mutation effects predicted from sequence co-variation.

Authors:  Thomas A Hopf; John B Ingraham; Frank J Poelwijk; Charlotta P I Schärfe; Michael Springer; Chris Sander; Debora S Marks
Journal:  Nat Biotechnol       Date:  2017-01-16       Impact factor: 54.908

4.  Cystic fibrosis: Identification and frequency of mutations in a mixed population from a low-income region in Northeastern Brazil.

Authors:  Laís R Mota; Lorena L de Castro; Tatiane da Anunciação Ferreira; Renata L L F de Lima; Maria Betânia P Toralles; Edna L Souza
Journal:  Pediatr Pulmonol       Date:  2018-05-04

Review 5.  Cystic fibrosis: a worldwide analysis of CFTR mutations--correlation with incidence data and application to screening.

Authors:  Joseph L Bobadilla; Milan Macek; Jason P Fine; Philip M Farrell
Journal:  Hum Mutat       Date:  2002-06       Impact factor: 4.878

6.  Cystic fibrosis in a southern Brazilian population: characteristics of 90% of the alleles.

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Journal:  Clin Genet       Date:  2007-09       Impact factor: 4.438

7.  Genetic basis of variable exon 9 skipping in cystic fibrosis transmembrane conductance regulator mRNA.

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Journal:  Nat Genet       Date:  1993-02       Impact factor: 38.330

8.  Three novel mutations in the CFTR gene identified in Galician patients.

Authors:  P Rana-Díez; C Colón; J R Alonso-Fernández; A Solar; J C Barros-Tizón; D Barros-Casas; J Sirvent; A Carracedo; F Barros
Journal:  J Cyst Fibros       Date:  2008-08-03       Impact factor: 5.482

9.  Mutations in the cystic fibrosis gene in patients with congenital absence of the vas deferens.

Authors:  M Chillón; T Casals; B Mercier; L Bassas; W Lissens; S Silber; M C Romey; J Ruiz-Romero; C Verlingue; M Claustres
Journal:  N Engl J Med       Date:  1995-06-01       Impact factor: 91.245

Review 10.  CFTR Modulators: Shedding Light on Precision Medicine for Cystic Fibrosis.

Authors:  Miquéias Lopes-Pacheco
Journal:  Front Pharmacol       Date:  2016-09-05       Impact factor: 5.810

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  1 in total

Review 1.  A comprehensive overview of the cystic fibrosis on the island of São Miguel (Azores, Portugal).

Authors:  Joana Rosa; Patrícia Gaspar-Silva; Paula Pacheco; Conceição Silva; Cláudia C Branco; Barbara S Vieira; Alexandra Carreiro; Juan Gonçalves; Luisa Mota-Vieira
Journal:  BMC Pediatr       Date:  2020-01-03       Impact factor: 2.125

  1 in total

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