Literature DB >> 8755664

Activation of endogenous deltaF508 cystic fibrosis transmembrane conductance regulator by phosphodiesterase inhibition.

T J Kelley1, L Al-Nakkash, C U Cotton, M L Drumm.   

Abstract

Many heterologously expressed mutants of the cystic fibrosis transmembrane conductance regulator (CFTR) exhibit residual chloride channel activity that can be stimulated by agonists of the adenylate cyclase/protein kinase A pathway. Because of clinical implications for cystic fibrosis of activating mutants in vivo, we are investigating whether deltaF508, the most common disease-associated CFTR mutation, can be activated in airway epithelial cells. We have found that, 36Cl- efflux can be stimulated 19-61% above baseline by beta-adrenoreceptor agonists and cGI-phosphodiesterase inhibitors in transformed nasal polyp (CF-T43) cells homozygous for the deltaF508 mutation. The increase in 36Cl- permeability is diminished by protein kinase A inhibitors and is not mediated by an increase in intracellular calcium concentrations. Preincubation of CF-T43 cells with CFTR anti-sense oligonucleotides prevented an increase in 36Cl- efflux in response to beta-agonist and phosphodiesterase inhibitor. Primary cells isolated from CF nasal polyps gave similar results. These data indicate that endogenous levels of deltaF508 protein can be stimulated to increase 36Cl- permeability in airway epithelial cells.

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Year:  1996        PMID: 8755664      PMCID: PMC507457          DOI: 10.1172/JCI118819

Source DB:  PubMed          Journal:  J Clin Invest        ISSN: 0021-9738            Impact factor:   14.808


  37 in total

1.  Altered chloride ion channel kinetics associated with the delta F508 cystic fibrosis mutation.

Authors:  W Dalemans; P Barbry; G Champigny; S Jallat; K Dott; D Dreyer; R G Crystal; A Pavirani; J P Lecocq; M Lazdunski
Journal:  Nature       Date:  1991 Dec 19-26       Impact factor: 49.962

2.  Chloride conductance expressed by delta F508 and other mutant CFTRs in Xenopus oocytes.

Authors:  M L Drumm; D J Wilkinson; L S Smit; R T Worrell; T V Strong; R A Frizzell; D C Dawson; F S Collins
Journal:  Science       Date:  1991-12-20       Impact factor: 47.728

3.  Antisense oligodeoxynucleotide to the cystic fibrosis gene inhibits anion transport in normal cultured sweat duct cells.

Authors:  E J Sorscher; K L Kirk; M L Weaver; T Jilling; J E Blalock; R D LeBoeuf
Journal:  Proc Natl Acad Sci U S A       Date:  1991-09-01       Impact factor: 11.205

4.  Processing of mutant cystic fibrosis transmembrane conductance regulator is temperature-sensitive.

Authors:  G M Denning; M P Anderson; J F Amara; J Marshall; A E Smith; M J Welsh
Journal:  Nature       Date:  1992-08-27       Impact factor: 49.962

5.  A1 adenosine-receptor antagonists activate chloride efflux from cystic fibrosis cells.

Authors:  O Eidelman; C Guay-Broder; P J van Galen; K A Jacobson; C Fox; R J Turner; Z I Cabantchik; H B Pollard
Journal:  Proc Natl Acad Sci U S A       Date:  1992-06-15       Impact factor: 11.205

6.  Antisense oligodeoxynucleotides to the cystic fibrosis transmembrane conductance regulator inhibit cAMP-activated but not calcium-activated chloride currents.

Authors:  J A Wagner; T V McDonald; P T Nghiem; A W Lowe; H Schulman; D C Gruenert; L Stryer; P Gardner
Journal:  Proc Natl Acad Sci U S A       Date:  1992-08-01       Impact factor: 11.205

7.  Regulation of Cl- channels in normal and cystic fibrosis airway epithelial cells by extracellular ATP.

Authors:  M J Stutts; T C Chinet; S J Mason; J M Fullton; L L Clarke; R C Boucher
Journal:  Proc Natl Acad Sci U S A       Date:  1992-03-01       Impact factor: 11.205

8.  Purification and functional reconstitution of the cystic fibrosis transmembrane conductance regulator (CFTR).

Authors:  C E Bear; C H Li; N Kartner; R J Bridges; T J Jensen; M Ramjeesingh; J R Riordan
Journal:  Cell       Date:  1992-02-21       Impact factor: 41.582

9.  GTP-binding proteins inhibit cAMP activation of chloride channels in cystic fibrosis airway epithelial cells.

Authors:  E M Schwiebert; N Kizer; D C Gruenert; B A Stanton
Journal:  Proc Natl Acad Sci U S A       Date:  1992-11-15       Impact factor: 11.205

10.  Regulation of the cystic fibrosis transmembrane conductance regulator Cl- channel by specific protein kinases and protein phosphatases.

Authors:  H A Berger; S M Travis; M J Welsh
Journal:  J Biol Chem       Date:  1993-01-25       Impact factor: 5.157

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  11 in total

1.  Anchored PDE4 regulates chloride conductance in wild-type and ΔF508-CFTR human airway epithelia.

Authors:  Elise Blanchard; Lorna Zlock; Anna Lao; Delphine Mika; Wan Namkung; Moses Xie; Colleen Scheitrum; Dieter C Gruenert; Alan S Verkman; Walter E Finkbeiner; Marco Conti; Wito Richter
Journal:  FASEB J       Date:  2013-11-07       Impact factor: 5.191

Review 2.  Strategies for correcting the delta F508 CFTR protein-folding defect.

Authors:  C R Brown; L Q Hong-Brown; W J Welch
Journal:  J Bioenerg Biomembr       Date:  1997-10       Impact factor: 2.945

Review 3.  Cystic fibrosis genetics: from molecular understanding to clinical application.

Authors:  Garry R Cutting
Journal:  Nat Rev Genet       Date:  2014-11-18       Impact factor: 53.242

Review 4.  Targeted therapy for cystic fibrosis: cystic fibrosis transmembrane conductance regulator mutation-specific pharmacologic strategies.

Authors:  Ronald C Rubenstein
Journal:  Mol Diagn Ther       Date:  2006       Impact factor: 4.074

5.  Failure of cAMP agonists to activate rescued deltaF508 CFTR in CFBE41o- airway epithelial monolayers.

Authors:  Zsuzsa Bebok; James F Collawn; John Wakefield; William Parker; Yao Li; Karoly Varga; Eric J Sorscher; J P Clancy
Journal:  J Physiol       Date:  2005-10-06       Impact factor: 5.182

6.  Deletion of phenylalanine 508 causes attenuated phosphorylation-dependent activation of CFTR chloride channels.

Authors:  F Wang; S Zeltwanger; S Hu; T C Hwang
Journal:  J Physiol       Date:  2000-05-01       Impact factor: 5.182

Review 7.  Changing the Paradigm - Treating the Basic Defect in Cystic Fibrosis.

Authors:  Lokesh Guglani
Journal:  Indian J Pediatr       Date:  2015-06-17       Impact factor: 1.967

8.  DeltaF508 CFTR processing correction and activity in polarized airway and non-airway cell monolayers.

Authors:  S M Rowe; L C Pyle; A Jurkevante; K Varga; J Collawn; P A Sloane; B Woodworth; M Mazur; J Fulton; L Fan; Y Li; J Fortenberry; E J Sorscher; J P Clancy
Journal:  Pulm Pharmacol Ther       Date:  2010-03-10       Impact factor: 3.410

9.  In vivo activation of the cystic fibrosis transmembrane conductance regulator mutant deltaF508 in murine nasal epithelium.

Authors:  T J Kelley; K Thomas; L J Milgram; M L Drumm
Journal:  Proc Natl Acad Sci U S A       Date:  1997-03-18       Impact factor: 11.205

10.  Role of oxygen availability in CFTR expression and function.

Authors:  Jennifer S Guimbellot; James A Fortenberry; Gene P Siegal; Bryan Moore; Hui Wen; Charles Venglarik; Yiu-Fai Chen; Suzanne Oparil; Eric J Sorscher; Jeong S Hong
Journal:  Am J Respir Cell Mol Biol       Date:  2008-05-12       Impact factor: 6.914

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