High-dose IgG therapy mitigates bile duct-targeted inflammation and obstruction in a mouse model of biliary atresia

Pediatr Res. 2014 Jul;76(1):72-80. doi: 10.1038/pr.2014.46. Epub 2014 Apr 11.

Abstract

Background: A proposed etiology of biliary atresia (BA) entails a virus-induced, progressive immune-mediated injury of the biliary system. Intravenous Ig (IVIg) has demonstrated clinical benefit in several inflammatory diseases. The aim of this study was to determine the therapeutic effects of high-dose IgG treatment in the rhesus rotavirus (RRV)-induced mouse model of BA.

Methods: Newborn mice were infected with RRV, and jaundiced mice were given high-dose IgG or albumin control. Survival, histology, direct bilirubin, liver immune cell subsets, and cytokine production were analyzed.

Results: There was no difference in overall survival between RRV-infected groups, however high-dose IgG resulted in decreased bilirubin, bile duct inflammation, and increased extrahepatic bile duct patency. High-dose IgG decreased vascular cell adhesion molecule-1, resulting in limited migration of immune cells to portal tracts. High-dose IgG significantly decreased CD4(+) T cell production of interleukin (IL)-2, interferon (IFN)-γ, and tumor necrosis factor (TNF)-α and CD8(+) T cell production of IFN-γ, as well as increased levels of regulatory T cells.

Conclusion: High-dose IgG therapy in murine BA dramatically decreased Th1 cell-mediated inflammation and biliary obstruction. This study lends support for consideration of IVIg clinical trials in infants with BA, to diminish the progressive intrahepatic bile duct injury.

Publication types

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

MeSH terms

  • Albumins / therapeutic use
  • Animals
  • Bile Ducts / drug effects*
  • Bile Ducts / pathology
  • Biliary Atresia / immunology*
  • Biliary Atresia / therapy*
  • Bilirubin / analysis
  • CD4-Positive T-Lymphocytes / cytology
  • CD8-Positive T-Lymphocytes / cytology
  • Disease Models, Animal
  • Immunoglobulin G / therapeutic use*
  • Immunoglobulins, Intravenous / therapeutic use
  • Inflammation / therapy*
  • Interferon-gamma / metabolism
  • Interleukin-2 / metabolism
  • Mice
  • Rotavirus
  • Tumor Necrosis Factor-alpha / metabolism
  • Vascular Cell Adhesion Molecule-1 / metabolism

Substances

  • Albumins
  • Immunoglobulin G
  • Immunoglobulins, Intravenous
  • Interleukin-2
  • Tumor Necrosis Factor-alpha
  • Vascular Cell Adhesion Molecule-1
  • Interferon-gamma
  • Bilirubin