Chronic β2AR stimulation limits CFTR activation in human airway epithelia

JCI Insight. 2018 Feb 22;3(4):e93029. doi: 10.1172/jci.insight.93029.

Abstract

Traditional pulmonary therapies for cystic fibrosis (CF) target the downstream effects of CF transmembrane conductance regulator (CFTR) dysfunction (the cause of CF). Use of one such therapy, β-adrenergic bronchodilators (such as albuterol), is nearly universal for airway clearance. Conversely, novel modulator therapies restore function to select mutant CFTR proteins, offering a disease-modifying treatment. Recent trials of modulators targeting F508del-CFTR, the most common CFTR mutation, suggest that chronic β-agonist use may undermine clinical modulator benefits. We therefore sought to understand the impact of chronic or excess β-agonist exposure on CFTR activation in human airway epithelium. The present studies demonstrate a greater than 60% reduction in both wild-type and modulator-corrected F508del-CFTR activation following chronic exposure to short- and long-acting β-agonists. This reduction was due to reduced cellular generation of cAMP downstream of the β-2 adrenergic receptor-G protein complex. Our results point towards a posttranscriptional reduction in adenylyl cyclase function as the mechanism of impaired CFTR activation produced by prolonged β-agonist exposure. β-Agonist-induced CFTR dysfunction was sufficient to abrogate VX809/VX770 modulation of F508del-CFTR in vitro. Understanding the clinical relevance of our observations is critical for CF patients using these drugs, and for investigators to inform future CFTR modulator drug trials.

Keywords: Cell Biology; Chloride channels; Cyclic nucleotides; Epithelial transport of ions and water; Pulmonology.

Publication types

  • Research Support, N.I.H., Extramural
  • Research Support, Non-U.S. Gov't

MeSH terms

  • Adrenergic beta-2 Receptor Agonists / pharmacology*
  • Adrenergic beta-2 Receptor Agonists / therapeutic use
  • Albuterol / pharmacology
  • Albuterol / therapeutic use
  • Aminophenols / pharmacology*
  • Aminophenols / therapeutic use
  • Aminopyridines / pharmacology*
  • Aminopyridines / therapeutic use
  • Benzodioxoles / pharmacology*
  • Benzodioxoles / therapeutic use
  • Cell Line
  • Cilia / drug effects
  • Cilia / pathology
  • Cyclic AMP / metabolism
  • Cystic Fibrosis / drug therapy*
  • Cystic Fibrosis / genetics
  • Cystic Fibrosis / pathology
  • Cystic Fibrosis Transmembrane Conductance Regulator / genetics
  • Cystic Fibrosis Transmembrane Conductance Regulator / metabolism*
  • Drug Interactions
  • Epithelial Cells / cytology
  • Epithelial Cells / drug effects
  • Epithelial Cells / pathology
  • Humans
  • Mutation
  • Quinolones / pharmacology*
  • Quinolones / therapeutic use
  • Respiratory Mucosa / cytology
  • Respiratory Mucosa / drug effects*
  • Respiratory Mucosa / pathology
  • Time Factors

Substances

  • Adrenergic beta-2 Receptor Agonists
  • Aminophenols
  • Aminopyridines
  • Benzodioxoles
  • CFTR protein, human
  • Quinolones
  • Cystic Fibrosis Transmembrane Conductance Regulator
  • ivacaftor
  • Cyclic AMP
  • lumacaftor
  • Albuterol