Different CFTR modulator combinations downregulate inflammation differently in cystic fibrosis

Elife. 2020 Mar 2:9:e54556. doi: 10.7554/eLife.54556.

Abstract

Previously, we showed that serum and monocytes from patients with CF exhibit an enhanced NLRP3-inflammasome signature with increased IL-18, IL-1β, caspase-1 activity and ASC speck release (Scambler et al. eLife 2019). Here we show that CFTR modulators down regulate this exaggerated proinflammatory response following LPS/ATP stimulation. In vitro application of ivacaftor/lumacaftor or ivacaftor/tezacaftor to CF monocytes showed a significant reduction in IL-18, whereas IL-1β was only reduced with ivacaftor/tezacaftor. Thirteen adults starting ivacaftor/lumacaftor and eight starting ivacaftor/tezacaftor were assessed over three months. Serum IL-18 and TNF decreased significantly with treatments, but IL-1β only declined following ivacaftor/tezacaftor. In (LPS/ATP-stimulated) PBMCs, IL-18/TNF/caspase-1 were all significantly decreased and IL-10 was increased with both combinations. Ivacaftor/tezacaftor alone showed a significant reduction in IL-1β and pro-IL-1β mRNA. This study demonstrates that these CFTR modulator combinations have potent anti-inflammatory properties, in addition to their ability to stimulate CFTR function, which could contribute to improved clinical outcomes.

Keywords: cystic fibrosis; human; immunology; inflammation; ivacaftor; lumacaftor; nlrp3 inflammasome; tezacaftor.

Publication types

  • Research Support, Non-U.S. Gov't

MeSH terms

  • Adult
  • Aminophenols / administration & dosage
  • Aminophenols / therapeutic use*
  • Aminopyridines / administration & dosage
  • Aminopyridines / therapeutic use*
  • Benzodioxoles / administration & dosage
  • Benzodioxoles / therapeutic use*
  • Cystic Fibrosis / drug therapy
  • Cystic Fibrosis / metabolism*
  • Cystic Fibrosis Transmembrane Conductance Regulator / drug effects*
  • Cystic Fibrosis Transmembrane Conductance Regulator / metabolism
  • Cytokines / metabolism
  • Down-Regulation
  • Drug Therapy, Combination
  • Female
  • Humans
  • Indoles / administration & dosage
  • Indoles / therapeutic use*
  • Inflammation / diet therapy
  • Inflammation / metabolism*
  • Interleukin-18 / blood
  • Interleukin-1beta / blood
  • Male
  • Monocytes / drug effects
  • Monocytes / metabolism
  • Quinolones / administration & dosage
  • Quinolones / therapeutic use*
  • Tumor Necrosis Factor-alpha / blood
  • Young Adult

Substances

  • Aminophenols
  • Aminopyridines
  • Benzodioxoles
  • CFTR protein, human
  • Cytokines
  • IL18 protein, human
  • Indoles
  • Interleukin-18
  • Interleukin-1beta
  • Quinolones
  • Tumor Necrosis Factor-alpha
  • tezacaftor
  • Cystic Fibrosis Transmembrane Conductance Regulator
  • ivacaftor
  • lumacaftor