Literature DB >> 10099982

The complex relationships between cystic fibrosis and congenital bilateral absence of the vas deferens: clinical, electrophysiological and genetic data.

G R Dohle1, H J Veeze, S E Overbeek, A M van den Ouweland, D J Halley, R F Weber, M F Niermeijer.   

Abstract

Congenital bilateral absence of the vas deferens (CBAVD) is found in 1-2% of infertile males and in most male cystic fibrosis (CF) patients. CF and some of the CBAVD cases were found to share the same genetic background. In this study, 21 males with CBAVD had extensive physical and laboratory testing for symptoms of CF. Possible defective cellular chloride transport was measured by interstitial current measurement of rectal suction biopsies. Cystic fibrosis transmembrane conductance regulator (CFTR) gene mutation analysis was performed for 10 common CFTR mutations. CF-related symptoms were found in six men. On laboratory testing slightly abnormal liver and pancreatic function was found in seven patients. The sweat test was found to be abnormal in four patients; interstitial current measurement showed defective chloride excretion in 11 patients. CFTR gene mutations were found in 66% of the patients: eight were compound heterozygotes; in six, only one common mutation could be detected. The 5T allele in one copy of intron 8 was found in four men. CBAVD appears to be a heterogeneous clinical and genetic condition. A CFTR gene mutation was found in both copies of the allele or interstitial current measurement showed defective chloride excretion in 14/21 cases. Genetic counselling is clearly indicated for couples seeking pregnancy through epididymal or testicular sperm aspiration and intracytoplasmic sperm injection.

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Year:  1999        PMID: 10099982     DOI: 10.1093/humrep/14.2.371

Source DB:  PubMed          Journal:  Hum Reprod        ISSN: 0268-1161            Impact factor:   6.918


  6 in total

Review 1.  The cystic fibrosis transmembrane regulator gene and male infertility.

Authors:  C Quinzii; C Castellani
Journal:  J Endocrinol Invest       Date:  2000-11       Impact factor: 4.256

Review 2.  Atypical cystic fibrosis--diagnostic and management dilemmas.

Authors:  Colin Wallis
Journal:  J R Soc Med       Date:  2003       Impact factor: 5.344

Review 3.  Functional modules, mutational load and human genetic disease.

Authors:  Norann A Zaghloul; Nicholas Katsanis
Journal:  Trends Genet       Date:  2010-03-11       Impact factor: 11.639

4.  Mutations in the cystic fibrosis transmembrane regulator gene and in vivo transepithelial potentials.

Authors:  Michael Wilschanski; Annie Dupuis; Lynda Ellis; Keith Jarvi; Julian Zielenski; Elizabeth Tullis; Sheelagh Martin; Mary Corey; Lap-Chee Tsui; Peter Durie
Journal:  Am J Respir Crit Care Med       Date:  2006-07-13       Impact factor: 21.405

5.  In vivo readout of CFTR function: ratiometric measurement of CFTR-dependent secretion by individual, identifiable human sweat glands.

Authors:  Jeffrey J Wine; Jessica E Char; Jonathan Chen; Hyung-Ju Cho; Colleen Dunn; Eric Frisbee; Nam Soo Joo; Carlos Milla; Sara E Modlin; Il-Ho Park; Ewart A C Thomas; Kim V Tran; Rohan Verma; Marlene H Wolfe
Journal:  PLoS One       Date:  2013-10-24       Impact factor: 3.240

6.  Unilateral Kidney Agenesis and other Kidney Anomalies in Infertile Men with Congenital Bilateral Absence of Vas deferens: A Cross-Sectional Study.

Authors:  Fattaneh Pahlavan; Fatemeh Niknejad; Hesamoddin Sajadi; Ahmad Vosough
Journal:  Int J Fertil Steril       Date:  2022-08-21
  6 in total

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