Next-Generation Sequencing for Screening Analysis of Cystic Fibrosis: Spectrum and Novel Variants in a South-Central Italian Cohort.

CFTR cystic fibrosis cystic fibrosis carriers cystic fibrosis transmembrane conductance regulator gene next-generation sequencing

Journal

Genes
ISSN: 2073-4425
Titre abrégé: Genes (Basel)
Pays: Switzerland
ID NLM: 101551097

Informations de publication

Date de publication:
11 08 2023
Historique:
received: 22 07 2023
revised: 06 08 2023
accepted: 08 08 2023
medline: 28 8 2023
pubmed: 26 8 2023
entrez: 26 8 2023
Statut: epublish

Résumé

The incidence of cystic fibrosis (CF) and the spectrum of cystic fibrosis transmembrane conductance regulator (CFTR) gene variants differ among geographic regions. Differences in CF carrier distribution are also reported among Italian regions. We described the spectrum of the CFTR variants observed in a large group of subjects belonging from central-southern Italy. We also provide a predictive evaluation of the novel variants identified. CFTR screening was performed in a south-central Italian cohort of 770 subjects. We adopted a next-generation sequencing (NGS) approach using the Devyser CFTR NGS kit on the Illumina MiSeq System coupled with Amplicon Suite data analysis. Bioinformatics evaluation of the impact of novel variants was described. Overall, the presence of at least one alternative allele in the CFTR gene was recorded for 23% of the subjects, with a carrier frequency of CF pathogenic variants of 1:12. The largest sub-group corresponded to the heterozygous carriers of a variant with a conflicting interpretation of pathogenicity. The common CFTR p.(Phe508del) pathogenic variants were identified in 37% of mutated subjects. Bioinformatics prediction supported a potential damaging effect for the three novel

Identifiants

pubmed: 37628659
pii: genes14081608
doi: 10.3390/genes14081608
pmc: PMC10454170
pii:
doi:

Substances chimiques

Cystic Fibrosis Transmembrane Conductance Regulator 126880-72-6

Types de publication

Journal Article

Langues

eng

Sous-ensembles de citation

IM

Références

Eur Respir J. 2020 Sep 10;56(3):
pubmed: 32398304
Eur Respir J. 2020 Sep 10;56(3):
pubmed: 32912926
BMC Med Genet. 2020 May 1;21(1):89
pubmed: 32357917
Cells. 2021 Oct 22;10(11):
pubmed: 34831067
Eur Respir J. 2014 Jan;43(1):20-3
pubmed: 24381320
Genet Med. 2016 Feb;18(2):145-51
pubmed: 26087173
Health Policy. 2008 Feb;85(2):133-47
pubmed: 17728003
J Cyst Fibros. 2010 Jan;9(1):29-35
pubmed: 19897426
Expert Rev Respir Med. 2018 May;12(5):415-426
pubmed: 29580110
J Med Screen. 1997;4(4):179-80
pubmed: 9494914
J Med Case Rep. 2014 Oct 10;8:339
pubmed: 25304080
Hum Mutat. 2020 Feb;41(2):375-386
pubmed: 31674704
Bioinformatics. 2019 Jun 1;35(11):1978-1980
pubmed: 30376034
Science. 1989 Sep 8;245(4922):1073-80
pubmed: 2570460
J Cyst Fibros. 2012 Jul;11(4):281-7
pubmed: 22425241
Proc Natl Acad Sci U S A. 2020 Jan 21;117(3):1621-1627
pubmed: 31882447
Protein Sci. 2021 Jan;30(1):60-69
pubmed: 32881105
Arch Pediatr. 2020 Feb;27 Suppl 1:eS8-eS12
pubmed: 32172939
JAMA. 2009 Dec 16;302(23):2573-9
pubmed: 20009057
Am J Hum Genet. 2017 Feb 2;100(2):267-280
pubmed: 28132688
Nat Methods. 2010 Apr;7(4):248-9
pubmed: 20354512
Genet Med. 2007 Nov;9(11):739-44
pubmed: 18007142

Auteurs

Elisa De Paolis (E)

Departmental Unit of Molecular and Genomic Diagnostics, Fondazione Policlinico Universitario A. Gemelli IRCCS, 00168 Rome, Italy.
Department of Basic Biotechnological Sciences, Intensivological and Perioperative Clinics, Catholic University of Sacred Heart, 00168 Rome, Italy.

Bruno Tilocca (B)

Department of Health Science, University "Magna Graecia" of Catanzaro, 88100 Catanzaro, Italy.

Carla Lombardi (C)

Department of Basic Biotechnological Sciences, Intensivological and Perioperative Clinics, Catholic University of Sacred Heart, 00168 Rome, Italy.

Maria De Bonis (M)

Departmental Unit of Molecular and Genomic Diagnostics, Fondazione Policlinico Universitario A. Gemelli IRCCS, 00168 Rome, Italy.

Paola Concolino (P)

Departmental Unit of Molecular and Genomic Diagnostics, Fondazione Policlinico Universitario A. Gemelli IRCCS, 00168 Rome, Italy.

Maria Elisabetta Onori (ME)

Departmental Unit of Molecular and Genomic Diagnostics, Fondazione Policlinico Universitario A. Gemelli IRCCS, 00168 Rome, Italy.

Claudio Ricciardi Tenore (C)

Departmental Unit of Molecular and Genomic Diagnostics, Fondazione Policlinico Universitario A. Gemelli IRCCS, 00168 Rome, Italy.

Alessia Perrucci (A)

Departmental Unit of Molecular and Genomic Diagnostics, Fondazione Policlinico Universitario A. Gemelli IRCCS, 00168 Rome, Italy.

Paola Roncada (P)

Department of Health Science, University "Magna Graecia" of Catanzaro, 88100 Catanzaro, Italy.

Ettore Capoluongo (E)

Department of Molecular Medicine and Medical Biotechnologies, University Federico II, 80138 Naples, Italy.
Department of Clinical Pathology and Genomics, Ospedale Cannizzaro, 95021 Catania, Italy.

Andrea Urbani (A)

Departmental Unit of Molecular and Genomic Diagnostics, Fondazione Policlinico Universitario A. Gemelli IRCCS, 00168 Rome, Italy.
Department of Basic Biotechnological Sciences, Intensivological and Perioperative Clinics, Catholic University of Sacred Heart, 00168 Rome, Italy.

Angelo Minucci (A)

Departmental Unit of Molecular and Genomic Diagnostics, Fondazione Policlinico Universitario A. Gemelli IRCCS, 00168 Rome, Italy.

Concetta Santonocito (C)

Departmental Unit of Molecular and Genomic Diagnostics, Fondazione Policlinico Universitario A. Gemelli IRCCS, 00168 Rome, Italy.
Department of Basic Biotechnological Sciences, Intensivological and Perioperative Clinics, Catholic University of Sacred Heart, 00168 Rome, Italy.

Articles similaires

Genome, Chloroplast Phylogeny Genetic Markers Base Composition High-Throughput Nucleotide Sequencing

[Redispensing of expensive oral anticancer medicines: a practical application].

Lisanne N van Merendonk, Kübra Akgöl, Bastiaan Nuijen
1.00
Humans Antineoplastic Agents Administration, Oral Drug Costs Counterfeit Drugs

Smoking Cessation and Incident Cardiovascular Disease.

Jun Hwan Cho, Seung Yong Shin, Hoseob Kim et al.
1.00
Humans Male Smoking Cessation Cardiovascular Diseases Female
Humans United States Aged Cross-Sectional Studies Medicare Part C

Classifications MeSH