Altered cGMP dynamics at the plasma membrane contribute to diarrhea in ulcerative colitis

Am J Pathol. 2015 Oct;185(10):2790-804. doi: 10.1016/j.ajpath.2015.06.007. Epub 2015 Aug 8.

Abstract

Ulcerative colitis (UC) belongs to inflammatory bowel disorders, a group of gastrointestinal disorders that can produce serious recurring diarrhea in affected patients. The mechanism for UC- and inflammatory bowel disorder-associated diarrhea is not well understood. The cystic fibrosis transmembrane-conductance regulator (CFTR) chloride channel plays an important role in fluid and water transport across the intestinal mucosa. CFTR channel function is regulated in a compartmentalized manner through the formation of CFTR-containing macromolecular complexes at the plasma membrane. In this study, we demonstrate the involvement of a novel macromolecular signaling pathway that causes diarrhea in UC. We found that a nitric oxide-producing enzyme, inducible nitric oxide synthase (iNOS), is overexpressed under the plasma membrane and generates compartmentalized cGMP in gut epithelia in UC. The scaffolding protein Na(+)/H(+) exchanger regulatory factor 2 (NHERF2) bridges iNOS with CFTR, forming CFTR-NHERF2-iNOS macromolecular complexes that potentiate CFTR channel function via the nitric oxide-cGMP pathway under inflammatory conditions both in vitro and in vivo. Potential disruption of these complexes in Nherf2(-/-) mice may render them more resistant to CFTR-mediated secretory diarrhea than Nherf2(+/+) mice in murine colitis models. Our study provides insight into the mechanism of pathophysiologic occurrence of diarrhea in UC and suggests that targeting CFTR and CFTR-containing macromolecular complexes will ameliorate diarrheal symptoms and improve conditions associated with inflammatory bowel disorders.

Publication types

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

MeSH terms

  • Animals
  • Cell Membrane / metabolism*
  • Cells, Cultured
  • Colitis, Ulcerative / genetics
  • Colitis, Ulcerative / metabolism*
  • Cyclic GMP / metabolism*
  • Cystic Fibrosis Transmembrane Conductance Regulator / metabolism
  • Diarrhea / metabolism*
  • Disease Models, Animal
  • Humans
  • Male
  • Mice, Inbred C57BL
  • Mice, Knockout
  • Phosphoproteins / metabolism
  • Signal Transduction / physiology
  • Sodium-Hydrogen Exchangers / metabolism

Substances

  • Phosphoproteins
  • Sodium-Hydrogen Exchangers
  • sodium-hydrogen exchanger regulatory factor
  • Cystic Fibrosis Transmembrane Conductance Regulator
  • Cyclic GMP