A new murine model to define the critical pathologic and therapeutic mediators of polymyositis

Arthritis Rheum. 2007 Apr;56(4):1304-14. doi: 10.1002/art.22521.

Abstract

Objective: To establish a new murine model of polymyositis (PM) for the understanding of its pathologic mechanisms and the development of new treatment strategies.

Methods: C protein-induced myositis (CIM) was induced by a single immunization of recombinant human skeletal C protein in C57BL/6 mice, as well as in CD4-depleted, CD8-depleted, and mutant mice as controls. Some mice were treated with high-dose intravenous immunoglobulin (IVIG) after disease induction. Muscle tissues were examined histologically.

Results: In mice with CIM, inflammation was confined to the skeletal muscles. Histologic examination revealed a common pathologic feature of CIM and PM, involving abundant infiltration of CD8 and perforin-expressing cells in the endomysial site of the injured muscle. Suppression of myositis was achieved by depletion of both CD4 and CD8 T cells. Despite the development of serum anti-C protein antibodies in wild-type mice, severe myositis was induced in mice deficient in B cells. Induction of myositis was suppressed in interleukin-1alpha/beta (IL-1alpha/beta)-null mutant mice, but not in tumor necrosis factor alpha (TNFalpha)-null mutant mice. Use of IVIG, a treatment with proven efficacy in PM, suppressed CIM in the subgroup of treated mice.

Conclusion: CIM mimics PM pathologically and clinically. Infiltration of CD8 T cells is the most likely mechanism of muscle injury, and IL-1, but not B cells or TNFalpha, is crucial in the development of CIM. IVIG has therapeutic effects in CIM, suggesting that the effects of IVIG are not mediated by suppression of antibody-mediated tissue injury. This murine model provides a useful tool for understanding the pathologic mechanisms of PM and for developing new treatment strategies.

Publication types

  • Research Support, Non-U.S. Gov't

MeSH terms

  • Animals
  • CD4-Positive T-Lymphocytes / immunology
  • CD4-Positive T-Lymphocytes / pathology
  • CD8 Antigens / metabolism
  • CD8-Positive T-Lymphocytes / immunology
  • CD8-Positive T-Lymphocytes / pathology
  • Carrier Proteins / immunology*
  • Disease Models, Animal*
  • Female
  • Gene Silencing
  • Humans
  • Immunoglobulins, Intravenous / therapeutic use*
  • Mice
  • Mice, Inbred C57BL
  • Mice, Inbred DBA
  • Mice, Knockout
  • Mice, Mutant Strains
  • Muscle, Skeletal / immunology
  • Muscle, Skeletal / metabolism
  • Muscle, Skeletal / pathology*
  • Polymyositis / immunology
  • Polymyositis / pathology*
  • Polymyositis / therapy*
  • Postural Balance / drug effects
  • Postural Balance / physiology
  • Recombinant Proteins
  • Running

Substances

  • CD8 Antigens
  • Carrier Proteins
  • Immunoglobulins, Intravenous
  • Recombinant Proteins
  • myosin-binding protein C