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Prenatal Ultrasound Suspicion of Cystic Fibrosis in a Multiethnic Population: Is Extensive CFTR Genotyping Needed?

In families without a Cystic Fibrosis (CF) history, fetal ultrasound bowel abnormalities can unexpectedly reveal the disease. Isolated or in association, the signs can be fetal bowel hyperechogenicity, intestinal loop dilatation and non-visualization of fetal gallbladder. In these cases, search for...

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Autores principales: Mekki, Chadia, Aissat, Abdel, Mirlesse, Véronique, Mayer Lacrosniere, Sophie, Eche, Elsa, Le Floch, Annick, Whalen, Sandra, Prud’Homme, Cecile, Remus, Christelle, Funalot, Benoit, Castaigne, Vanina, Fanen, Pascale, de Becdelièvre, Alix
Formato: Online Artículo Texto
Lenguaje:English
Publicado: MDPI 2021
Materias:
Acceso en línea:https://www.ncbi.nlm.nih.gov/pmc/articles/PMC8145404/
https://www.ncbi.nlm.nih.gov/pubmed/33946859
http://dx.doi.org/10.3390/genes12050670
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author Mekki, Chadia
Aissat, Abdel
Mirlesse, Véronique
Mayer Lacrosniere, Sophie
Eche, Elsa
Le Floch, Annick
Whalen, Sandra
Prud’Homme, Cecile
Remus, Christelle
Funalot, Benoit
Castaigne, Vanina
Fanen, Pascale
de Becdelièvre, Alix
author_facet Mekki, Chadia
Aissat, Abdel
Mirlesse, Véronique
Mayer Lacrosniere, Sophie
Eche, Elsa
Le Floch, Annick
Whalen, Sandra
Prud’Homme, Cecile
Remus, Christelle
Funalot, Benoit
Castaigne, Vanina
Fanen, Pascale
de Becdelièvre, Alix
author_sort Mekki, Chadia
collection PubMed
description In families without a Cystic Fibrosis (CF) history, fetal ultrasound bowel abnormalities can unexpectedly reveal the disease. Isolated or in association, the signs can be fetal bowel hyperechogenicity, intestinal loop dilatation and non-visualization of fetal gallbladder. In these cases, search for CF transmembrane conductance regulator (CFTR) gene mutations is part of the recommended diagnostic practices, with a search for frequent mutations according to ethnicity, and, in case of the triad of signs, with an exhaustive study of the gene. However, the molecular diagnosis remains a challenge in populations without well-known frequent pathogenic variants. We present a multiethnic cohort of 108 pregnancies with fetal bowel abnormalities in which the parents benefited from an exhaustive study of the CFTR gene. We describe the new homozygous p.Cys1410* mutation in a fetus of African origin. We did not observe the most frequent p.Phe508del mutation in our cohort but evidenced variants undetected by our frequent mutations kit. Thanks to the progress of sequencing techniques and despite the difficulties of interpretation occasionally encountered, we discuss the need to carry out a comprehensive CFTR study in all patients in case of fetal bowel abnormalities.
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spelling pubmed-81454042021-05-26 Prenatal Ultrasound Suspicion of Cystic Fibrosis in a Multiethnic Population: Is Extensive CFTR Genotyping Needed? Mekki, Chadia Aissat, Abdel Mirlesse, Véronique Mayer Lacrosniere, Sophie Eche, Elsa Le Floch, Annick Whalen, Sandra Prud’Homme, Cecile Remus, Christelle Funalot, Benoit Castaigne, Vanina Fanen, Pascale de Becdelièvre, Alix Genes (Basel) Article In families without a Cystic Fibrosis (CF) history, fetal ultrasound bowel abnormalities can unexpectedly reveal the disease. Isolated or in association, the signs can be fetal bowel hyperechogenicity, intestinal loop dilatation and non-visualization of fetal gallbladder. In these cases, search for CF transmembrane conductance regulator (CFTR) gene mutations is part of the recommended diagnostic practices, with a search for frequent mutations according to ethnicity, and, in case of the triad of signs, with an exhaustive study of the gene. However, the molecular diagnosis remains a challenge in populations without well-known frequent pathogenic variants. We present a multiethnic cohort of 108 pregnancies with fetal bowel abnormalities in which the parents benefited from an exhaustive study of the CFTR gene. We describe the new homozygous p.Cys1410* mutation in a fetus of African origin. We did not observe the most frequent p.Phe508del mutation in our cohort but evidenced variants undetected by our frequent mutations kit. Thanks to the progress of sequencing techniques and despite the difficulties of interpretation occasionally encountered, we discuss the need to carry out a comprehensive CFTR study in all patients in case of fetal bowel abnormalities. MDPI 2021-04-29 /pmc/articles/PMC8145404/ /pubmed/33946859 http://dx.doi.org/10.3390/genes12050670 Text en © 2021 by the authors. https://creativecommons.org/licenses/by/4.0/Licensee MDPI, Basel, Switzerland. This article is an open access article distributed under the terms and conditions of the Creative Commons Attribution (CC BY) license (https://creativecommons.org/licenses/by/4.0/).
spellingShingle Article
Mekki, Chadia
Aissat, Abdel
Mirlesse, Véronique
Mayer Lacrosniere, Sophie
Eche, Elsa
Le Floch, Annick
Whalen, Sandra
Prud’Homme, Cecile
Remus, Christelle
Funalot, Benoit
Castaigne, Vanina
Fanen, Pascale
de Becdelièvre, Alix
Prenatal Ultrasound Suspicion of Cystic Fibrosis in a Multiethnic Population: Is Extensive CFTR Genotyping Needed?
title Prenatal Ultrasound Suspicion of Cystic Fibrosis in a Multiethnic Population: Is Extensive CFTR Genotyping Needed?
title_full Prenatal Ultrasound Suspicion of Cystic Fibrosis in a Multiethnic Population: Is Extensive CFTR Genotyping Needed?
title_fullStr Prenatal Ultrasound Suspicion of Cystic Fibrosis in a Multiethnic Population: Is Extensive CFTR Genotyping Needed?
title_full_unstemmed Prenatal Ultrasound Suspicion of Cystic Fibrosis in a Multiethnic Population: Is Extensive CFTR Genotyping Needed?
title_short Prenatal Ultrasound Suspicion of Cystic Fibrosis in a Multiethnic Population: Is Extensive CFTR Genotyping Needed?
title_sort prenatal ultrasound suspicion of cystic fibrosis in a multiethnic population: is extensive cftr genotyping needed?
topic Article
url https://www.ncbi.nlm.nih.gov/pmc/articles/PMC8145404/
https://www.ncbi.nlm.nih.gov/pubmed/33946859
http://dx.doi.org/10.3390/genes12050670
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