Abstract
Secretory diarrhea is the leading cause of infant death in developing countries and a major cause of morbidity in adults. The cystic fibrosis transmembrane conductance regulator (CFTR) protein is required for fluid secretion in the intestine and airways and, when defective, causes the lethal genetic disease cystic fibrosis. We screened 50,000 chemically diverse compounds for inhibition of cAMP/flavone-stimulated Cl(-) transport in epithelial cells expressing CFTR. Six CFTR inhibitors of the 2-thioxo-4-thiazolidinone chemical class were identified. The most potent compound discovered by screening of structural analogs, CFTR(inh)-172, reversibly inhibited CFTR short-circuit current in less than 2 minutes in a voltage-independent manner with K(I) approximately 300 nM. CFTR(inh)-172 was nontoxic at high concentrations in cell culture and mouse models. At concentrations fully inhibiting CFTR, CFTR(inh)-172 did not prevent elevation of cellular cAMP or inhibit non-CFTR Cl(-) channels, multidrug resistance protein-1 (MDR-1), ATP-sensitive K(+) channels, or a series of other transporters. A single intraperitoneal injection of CFTR(inh)-172 (250 micro g/kg) in mice reduced by more than 90% cholera toxin-induced fluid secretion in the small intestine over 6 hours. Thiazolidinone CFTR inhibitors may be useful in developing large-animal models of cystic fibrosis and in reducing intestinal fluid loss in cholera and other secretory diarrheas.
MeSH Terms
Amino Acid Substitution
Animals
Cell Membrane/drug effects,physiology
Cholera Toxin/toxicity
Colforsin/pharmacology
Cystic Fibrosis Transmembrane Conductance Regulator/antagonists & inhibitors,drug effects,genetics
Diarrhea/etiology,prevention & control
Humans
Ileum/drug effects,physiology
Mice
Muscle, Smooth/drug effects,physiology
Mutagenesis, Site-Directed
Patch-Clamp Techniques
Rats
Rats, Inbred F344
Recombinant Proteins/antagonists & inhibitors
Thiazoles/pharmacology
Chemicals
CFTR protein, human
Recombinant Proteins
Thiazoles
Cystic Fibrosis Transmembrane Conductance Regulator
Colforsin
Cholera Toxin
Authors & Affiliations
7 authors, click to expand affiliations / ORCID
Ma Tonghui
Department of Medicine, Cardiovascular Research Institute, University of California, San Francisco, San Francisco, California 94143-0521, USA.
Thiagarajah Jay R
Yang Hong
Sonawane Nitin D
Folli Chiara
Galietta Luis J V
Verkman A S
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