Literature DB >> 31406621

Homozygous Deletion of the CFTR Gene Caused by Interstitial Maternal Isodisomy in a Peruvian Child with Cystic Fibrosis.

Flor Vásquez Sotomayor1, Hugo Hernán Abarca-Barriga1,2.   

Abstract

We report the first case in Peru of cystic fibrosis caused by a homozygous deletion of the cystic fibrosis transmembrane conductance regulator ( CFTR ) gene. A 10-month-old child who presented with meconium ileus and pancreatic insufficiency was tested for cystic fibrosis. Both parents of the child are of Peruvian background, are nonconsanguineous, and have no personal or family history of the disease. Chromosome microarray analysis revealed a homozygous deletion of the CFTR gene on chromosome 7 (7q31.2) within a maternally derived 12.8-Mb region of loss of heterozygosity with deletion of a region that includes the CFTR gene. Parental testing confirmed this finding. This case highlights the great importance of molecular testing and the study of chromosomal rearrangements in reaching a correct diagnosis and providing proper genetic counseling to the affected families.

Entities:  

Keywords:  CFTR gene ; chromosome microarray analysis; copy number variation; cystic fibrosis; loss of heterozygosity; uniparental disomy

Year:  2019        PMID: 31406621      PMCID: PMC6688885          DOI: 10.1055/s-0039-1678682

Source DB:  PubMed          Journal:  J Pediatr Genet        ISSN: 2146-460X


  23 in total

1.  Rapid detection of CFTR gene rearrangements impacts on genetic counselling in cystic fibrosis.

Authors:  F Niel; J Martin; F Dastot-Le Moal; B Costes; B Boissier; V Delattre; M Goossens; E Girodon
Journal:  J Med Genet       Date:  2004-11       Impact factor: 6.318

2.  Identification and characterization of three large deletions and a deletion/polymorphism in the CFTR gene.

Authors:  F Chevalier-Porst; G Souche; D Bozon
Journal:  Hum Mutat       Date:  2005-05       Impact factor: 4.878

3.  Cystic fibrosis.

Authors:  Steven M Rowe; Stacey Miller; Eric J Sorscher
Journal:  N Engl J Med       Date:  2005-05-12       Impact factor: 91.245

4.  Novel and recurrent rearrangements in the CFTR gene: clinical and laboratory implications for cystic fibrosis screening.

Authors:  Feras M Hantash; Joy B Redman; Kelsey Starn; Ben Anderson; Arlene Buller; Matthew J McGinniss; Franklin Quan; Mei Peng; Weimin Sun; Charles M Strom
Journal:  Hum Genet       Date:  2005-12-17       Impact factor: 4.132

5.  Detection of exon deletions within an entire gene (CFTR) by relative quantification on the LightCycler.

Authors:  Mircea Schneider; Franziska Joncourt; Javier Sanz; Thomas von Känel; Sabina Gallati
Journal:  Clin Chem       Date:  2006-09-21       Impact factor: 8.327

6.  CFTR gene analysis in Latin American CF patients: heterogeneous origin and distribution of mutations across the continent.

Authors:  Martín M Pérez; María Cecilia Luna; Omar H Pivetta; Genoveva Keyeux
Journal:  J Cyst Fibros       Date:  2006-09-11       Impact factor: 5.482

Review 7.  Complex and segmental uniparental disomy (UPD): review and lessons from rare chromosomal complements.

Authors:  D Kotzot
Journal:  J Med Genet       Date:  2001-08       Impact factor: 6.318

8.  Frequency of large CFTR gene rearrangements in Italian CF patients.

Authors:  Cristina Bombieri; Alberto Bonizzato; Carlo Castellani; Baroukh M Assael; Pier Franco Pignatti
Journal:  Eur J Hum Genet       Date:  2005-05       Impact factor: 4.246

Review 9.  Cystic fibrosis: molecular biology and therapeutic implications.

Authors:  F S Collins
Journal:  Science       Date:  1992-05-08       Impact factor: 47.728

10.  Genomic rearrangements in the CFTR gene: extensive allelic heterogeneity and diverse mutational mechanisms.

Authors:  Marie-Pierre Audrézet; Jian-Min Chen; Odile Raguénès; Nadia Chuzhanova; Karine Giteau; Cédric Le Maréchal; Isabelle Quéré; David N Cooper; Claude Férec
Journal:  Hum Mutat       Date:  2004-04       Impact factor: 4.878

View more

北京卡尤迪生物科技股份有限公司 © 2022-2023.