Development of IgA nephropathy-like glomerulonephritis associated with Wiskott-Aldrich syndrome protein deficiency

Clin Immunol. 2012 Feb;142(2):160-6. doi: 10.1016/j.clim.2011.10.001. Epub 2011 Oct 19.

Abstract

Wiskott-Aldrich syndrome (WAS) is a rare X-linked disorder caused by mutations in the WAS gene. Glomerulonephritis is a frequent complication, however, histopathological data from affected patients is scarce because the thrombocytopenia that affects most patients is a contraindication to renal biopsies. We found that WASp-deficient mice develop proliferative glomerulonephritis reminiscent of human IgA nephropathy (IgAN). We examined whether increased aberrant IgA production is associated with the development of glomerulonephritis in WASp-deficient mice. Serum IgA and IgA production by splenic B cells was increased in WASp-deficient mice compared to wild-type (WT) mice. A lectin-binding study revealed a reduced ratio of sialylated and galactosylated IgA in the sera from old WASp-deficient mice. Circulating IgA-containing immune complexes showed significantly higher titers in WASp-deficient mice compared to WT mice. These results indicate that the increased IgA production and aberrant glycosylation of IgA may be critically involved in the pathogenesis of glomerulonephritis in WAS.

Publication types

  • Comparative Study
  • Research Support, N.I.H., Intramural

MeSH terms

  • Animals
  • B-Lymphocytes / immunology
  • Disease Models, Animal
  • Glomerulonephritis, IGA / immunology*
  • Glomerulonephritis, IGA / metabolism
  • Glomerulonephritis, IGA / pathology
  • Glycosylation
  • Humans
  • Immunoglobulin A / blood
  • Immunoglobulin A / metabolism*
  • Mice
  • Mice, Knockout
  • Spleen / immunology
  • Thrombocytopenia / metabolism
  • Wiskott-Aldrich Syndrome / immunology*
  • Wiskott-Aldrich Syndrome Protein / deficiency*

Substances

  • Immunoglobulin A
  • Was protein, mouse
  • Wiskott-Aldrich Syndrome Protein