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L138ins Variant of the CFTR Gene in Russian Infertile Men
(1) Introduction: Pathogenic variants in the CFTR (Cystic Fibrosis Transmembrane conductance Regulator, OMIM: 602421) gene cause Cystic Fibrosis (CF, OMIM: 219700) and CF-related disorders (CF-RD), often accompanied by obstructive azoospermia due to congenital bilateral aplasia of vas deferens (CBAV...
Autores principales: | , , , , , , , , , , |
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Formato: | Online Artículo Texto |
Lenguaje: | English |
Publicado: |
MDPI
2023
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Materias: | |
Acceso en línea: | https://www.ncbi.nlm.nih.gov/pmc/articles/PMC10379041/ https://www.ncbi.nlm.nih.gov/pubmed/37510311 http://dx.doi.org/10.3390/genes14071407 |
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author | Chernykh, Vyacheslav Sorokina, Tatyana Sedova, Anna Shtaut, Maria Solovova, Olga Marnat, Ekaterina Adyan, Tagui Beskorovaynaya, Tatyana Stepanova, Anna Shchagina, Olga Polyakov, Aleksandr |
author_facet | Chernykh, Vyacheslav Sorokina, Tatyana Sedova, Anna Shtaut, Maria Solovova, Olga Marnat, Ekaterina Adyan, Tagui Beskorovaynaya, Tatyana Stepanova, Anna Shchagina, Olga Polyakov, Aleksandr |
author_sort | Chernykh, Vyacheslav |
collection | PubMed |
description | (1) Introduction: Pathogenic variants in the CFTR (Cystic Fibrosis Transmembrane conductance Regulator, OMIM: 602421) gene cause Cystic Fibrosis (CF, OMIM: 219700) and CF-related disorders (CF-RD), often accompanied by obstructive azoospermia due to congenital bilateral aplasia of vas deferens (CBAVD, OMIM: 277180) in male patients. The L138ins (c.413_415dup; p. (Leu138dup)) is a mild variant in the CFTR gene that is relatively common among CF-patients in Slavic populations. The frequency of this variant in Russian infertile men has not been sufficiently studied; (2) Materials and Methods: The sample consisted of 6033 Russian infertile men. The patients were tested for 22 common in Russian populations pathogenic variants of the CFTR gene and the IVS9Tn-polymorphic locus of the intron 9. Molecular-genetic studies were performed using amplified fragment length polymorphism (AFLP-PCR), multiplex ligation-dependent probe amplification (MLPA), and nested PCR (for analysis of the IVS9Tn-polymorphic locus); (3) Results: Pathogenic variants in the CFTR were detected in 3.9% of patients. The most frequent variants were F508del and CFTRdele2.3(21kb), accounted for 61.0% and 7.1% of detected variants, respectively. The L138ins variant was detected in 17 (0.28%) individuals: one of them was homozygous, 10 patients were heterozygous, and 6 patients were compound-heterozygous (F508del/L138ins, n = 4; L138ins/N1303K, n = 1; L138ins/5T, n = 1). Two pathogenic CF-causing variants in the CFTR gene were detected in 8 patients, including 7 compound heterozygous (F508del/L138ins, n = 4; F508del/N1303K, n = 1; 2184insA/E92K, n = 1; 3849+10kbC>T/E92K, n = 1) and one homozygous (L138ins/L138ins). The L138ins variant was found in 7 out of 16 (43.75%) chromosomes in six of these patients. The most common pathogenic variant, F508del, was identified in five out of them, in 5 of 16 (31.25%) chromosomes. The allele frequency (AF) of the L138ins variant in the sample has been found to be 0.0014.; (4) Conclusions: The L138ins variant of the CFTR gene is the third most common variant after F508del and CFTRdele2.3(kb) among Russian infertile men. |
format | Online Article Text |
id | pubmed-10379041 |
institution | National Center for Biotechnology Information |
language | English |
publishDate | 2023 |
publisher | MDPI |
record_format | MEDLINE/PubMed |
spelling | pubmed-103790412023-07-29 L138ins Variant of the CFTR Gene in Russian Infertile Men Chernykh, Vyacheslav Sorokina, Tatyana Sedova, Anna Shtaut, Maria Solovova, Olga Marnat, Ekaterina Adyan, Tagui Beskorovaynaya, Tatyana Stepanova, Anna Shchagina, Olga Polyakov, Aleksandr Genes (Basel) Article (1) Introduction: Pathogenic variants in the CFTR (Cystic Fibrosis Transmembrane conductance Regulator, OMIM: 602421) gene cause Cystic Fibrosis (CF, OMIM: 219700) and CF-related disorders (CF-RD), often accompanied by obstructive azoospermia due to congenital bilateral aplasia of vas deferens (CBAVD, OMIM: 277180) in male patients. The L138ins (c.413_415dup; p. (Leu138dup)) is a mild variant in the CFTR gene that is relatively common among CF-patients in Slavic populations. The frequency of this variant in Russian infertile men has not been sufficiently studied; (2) Materials and Methods: The sample consisted of 6033 Russian infertile men. The patients were tested for 22 common in Russian populations pathogenic variants of the CFTR gene and the IVS9Tn-polymorphic locus of the intron 9. Molecular-genetic studies were performed using amplified fragment length polymorphism (AFLP-PCR), multiplex ligation-dependent probe amplification (MLPA), and nested PCR (for analysis of the IVS9Tn-polymorphic locus); (3) Results: Pathogenic variants in the CFTR were detected in 3.9% of patients. The most frequent variants were F508del and CFTRdele2.3(21kb), accounted for 61.0% and 7.1% of detected variants, respectively. The L138ins variant was detected in 17 (0.28%) individuals: one of them was homozygous, 10 patients were heterozygous, and 6 patients were compound-heterozygous (F508del/L138ins, n = 4; L138ins/N1303K, n = 1; L138ins/5T, n = 1). Two pathogenic CF-causing variants in the CFTR gene were detected in 8 patients, including 7 compound heterozygous (F508del/L138ins, n = 4; F508del/N1303K, n = 1; 2184insA/E92K, n = 1; 3849+10kbC>T/E92K, n = 1) and one homozygous (L138ins/L138ins). The L138ins variant was found in 7 out of 16 (43.75%) chromosomes in six of these patients. The most common pathogenic variant, F508del, was identified in five out of them, in 5 of 16 (31.25%) chromosomes. The allele frequency (AF) of the L138ins variant in the sample has been found to be 0.0014.; (4) Conclusions: The L138ins variant of the CFTR gene is the third most common variant after F508del and CFTRdele2.3(kb) among Russian infertile men. MDPI 2023-07-07 /pmc/articles/PMC10379041/ /pubmed/37510311 http://dx.doi.org/10.3390/genes14071407 Text en © 2023 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 Chernykh, Vyacheslav Sorokina, Tatyana Sedova, Anna Shtaut, Maria Solovova, Olga Marnat, Ekaterina Adyan, Tagui Beskorovaynaya, Tatyana Stepanova, Anna Shchagina, Olga Polyakov, Aleksandr L138ins Variant of the CFTR Gene in Russian Infertile Men |
title | L138ins Variant of the CFTR Gene in Russian Infertile Men |
title_full | L138ins Variant of the CFTR Gene in Russian Infertile Men |
title_fullStr | L138ins Variant of the CFTR Gene in Russian Infertile Men |
title_full_unstemmed | L138ins Variant of the CFTR Gene in Russian Infertile Men |
title_short | L138ins Variant of the CFTR Gene in Russian Infertile Men |
title_sort | l138ins variant of the cftr gene in russian infertile men |
topic | Article |
url | https://www.ncbi.nlm.nih.gov/pmc/articles/PMC10379041/ https://www.ncbi.nlm.nih.gov/pubmed/37510311 http://dx.doi.org/10.3390/genes14071407 |
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