Progression of Alport Kidney Disease in Col4a3 Knock Out Mice Is Independent of Sex or Macrophage Depletion by Clodronate Treatment

PLoS One. 2015 Nov 10;10(11):e0141231. doi: 10.1371/journal.pone.0141231. eCollection 2015.

Abstract

Alport syndrome is a genetic disease of collagen IV (α3, 4, 5) resulting in renal failure. This study was designed to investigate sex-phenotype correlations and evaluate the contribution of macrophage infiltration to disease progression using Col4a3 knock out (Col4a3KO) mice, an established genetic model of autosomal recessive Alport syndrome. No sex differences in the evolution of body mass loss, renal pathology, biomarkers of tubular damage KIM-1 and NGAL, or deterioration of kidney function were observed during the life span of Col4a3KO mice. These findings confirm that, similar to human autosomal recessive Alport syndrome, female and male Col4a3KO mice develop renal failure at the same age and with similar severity. The specific contribution of macrophage infiltration to Alport disease, one of the prominent features of the disease in human and Col4a3KO mice, remains unknown. This study shows that depletion of kidney macrophages in Col4a3KO male mice by administration of clodronate liposomes, prior to clinical onset of disease and throughout the study period, does not protect the mice from renal failure and interstitial fibrosis, nor delay disease progression. These results suggest that therapy targeting macrophage recruitment to kidney is unlikely to be effective as treatment of Alport syndrome.

Publication types

  • Research Support, Non-U.S. Gov't

MeSH terms

  • Animals
  • Apoptosis
  • Autoantigens / genetics
  • Clodronic Acid / administration & dosage
  • Clodronic Acid / pharmacology
  • Clodronic Acid / therapeutic use*
  • Collagen Type IV / deficiency*
  • Collagen Type IV / genetics
  • Disease Progression
  • Drug Evaluation, Preclinical
  • Female
  • Kidney / pathology
  • Kidney Failure, Chronic / etiology
  • Liposomes
  • Macrophages / drug effects*
  • Macrophages / pathology
  • Male
  • Mice
  • Mice, 129 Strain
  • Mice, Knockout
  • Models, Animal
  • Nephritis, Hereditary / drug therapy
  • Nephritis, Hereditary / genetics
  • Nephritis, Hereditary / immunology
  • Nephritis, Hereditary / physiopathology*
  • Sex Characteristics

Substances

  • Autoantigens
  • Collagen Type IV
  • Liposomes
  • type IV collagen alpha3 chain
  • Clodronic Acid

Grants and funding

Funding for this work was provided by the Novartis Institute of Biomedical Research (NIBR). The funder provided support in the form of salaries and research materials for authors (MK, AP, NS, DK, BG, BK, DW, WD and IK), but did not have any additional role in the study design, data collection and analysis, decision to publish, or preparation of the manuscript. The specific roles of these authors are articulated in the ‘author contributions’ section.