A Cryptosporidium PI(4)K inhibitor is a drug candidate for cryptosporidiosis

Nature. 2017 Jun 15;546(7658):376-380. doi: 10.1038/nature22337. Epub 2017 May 31.

Abstract

Diarrhoeal disease is responsible for 8.6% of global child mortality. Recent epidemiological studies found the protozoan parasite Cryptosporidium to be a leading cause of paediatric diarrhoea, with particularly grave impact on infants and immunocompromised individuals. There is neither a vaccine nor an effective treatment. Here we establish a drug discovery process built on scalable phenotypic assays and mouse models that take advantage of transgenic parasites. Screening a library of compounds with anti-parasitic activity, we identify pyrazolopyridines as inhibitors of Cryptosporidium parvum and Cryptosporidium hominis. Oral treatment with the pyrazolopyridine KDU731 results in a potent reduction in intestinal infection of immunocompromised mice. Treatment also leads to rapid resolution of diarrhoea and dehydration in neonatal calves, a clinical model of cryptosporidiosis that closely resembles human infection. Our results suggest that the Cryptosporidium lipid kinase PI(4)K (phosphatidylinositol-4-OH kinase) is a target for pyrazolopyridines and that KDU731 warrants further preclinical evaluation as a drug candidate for the treatment of cryptosporidiosis.

Publication types

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

MeSH terms

  • 1-Phosphatidylinositol 4-Kinase / antagonists & inhibitors*
  • Animals
  • Animals, Newborn
  • Cattle
  • Cell Line, Tumor
  • Cryptosporidiosis / drug therapy*
  • Cryptosporidiosis / parasitology*
  • Cryptosporidium / drug effects*
  • Cryptosporidium / enzymology*
  • Disease Models, Animal
  • Female
  • Humans
  • Immunocompromised Host
  • Interferon-gamma / deficiency
  • Interferon-gamma / genetics
  • Male
  • Mice
  • Mice, Knockout
  • Pyrazoles / chemistry
  • Pyrazoles / pharmacokinetics
  • Pyrazoles / pharmacology*
  • Pyridines / chemistry
  • Pyridines / pharmacokinetics
  • Pyridines / pharmacology*
  • Rats
  • Rats, Wistar

Substances

  • KDU731
  • Pyrazoles
  • Pyridines
  • pyrazolopyridine
  • Interferon-gamma
  • 1-Phosphatidylinositol 4-Kinase