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PMID: 7529962 Published · ppublish English Journal Article Research Support, Non-U.S. Gov't

Is congenital bilateral absence of vas deferens a primary form of cystic fibrosis? Analyses of the CFTR gene in 67 patients.

American journal of human genetics ·Vol. 56 ·No. 1 ·1995-01-00 ·Pages 272-7

Mercier B, Verlingue C, Lissens W, Silber SJ, Novelli G, Bonduelle M, Audrézet MP, Férec C

Abstract

Congenital bilateral absence of the vas deferens (CBAVD) is an important cause of sterility in men. Although the genetic basis of this condition is still unclear, it has been shown recently that some of these patients carry mutations in their cystic fibrosis transmembrane conductance regulator (CFTR) genes. To extend this observation, we have analyzed the entire coding sequence of the CFTR gene in a cohort of 67 men with CBAVD, who are otherwise healthy. We have identified four novel missense mutations (A800G, G149R, R258G, and E193K). We have shown that 42% of subjects were carriers of one CFTR allele and that 24% are compound heterozygous for CFTR alleles. Thus, we have been unable to identify 76% of these patients as carrying two CFTR mutations. Furthermore, we have described the segregation of CFTR haplotypes in the family of one CBAVD male; in this family are two male siblings, with identical CFTR loci but displaying different phenotypes, one of them being fertile and the other sterile. The data presented in this family, indicating a discordance between the CBAVD phenotype and a marked carrier (delta F508) chromosome, support the involvement of another gene(s), in the etiology of CBAVD.

Related Genes
MeSH Terms
Alleles Base Sequence Cohort Studies Cystic Fibrosis/genetics Cystic Fibrosis Transmembrane Conductance Regulator Epistasis, Genetic Female Heterozygote Humans Infertility, Male/genetics Male Membrane Proteins/genetics Molecular Sequence Data Pedigree Point Mutation Vas Deferens/abnormalities
Chemicals
CFTR protein, human Membrane Proteins Cystic Fibrosis Transmembrane Conductance Regulator
Authors & Affiliations
8 authors, click to expand affiliations / ORCID
Mercier B
Centre de Biogénétique C.D.T.S., Brest, France.
Verlingue C
Lissens W
Silber S J
Novelli G
Bonduelle M
Audrézet M P
Férec C
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Article Info
Journal
American journal of human genetics
Abbr.
Am J Hum Genet
ISSN
0002-9297
Published
1995-01-00
Pages
272-7
Language
English
Region
United States
NLM ID
0370475
PMCID
PMC1801292
Subset
IM
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