Immunization with desmoglein 3 induces non-pathogenic autoantibodies in mice

PLoS One. 2021 Nov 3;16(11):e0259586. doi: 10.1371/journal.pone.0259586. eCollection 2021.

Abstract

Background: Pemphigus vulgaris (PV) is a rare autoimmune blistering disease characterized by the development of autoantibodies targeting desmoglein (Dsg) 3, but also against Dsg1 in mucocutaneous disease. Given that existing PV animal models only recapitulate aspects of the disease, we aimed to establish a more comprehensive disease model based on the immunization of mice with PV autoantigen(s).

Methods: The following immunization strategies were tested: (i) C57Bl/6J, B6.SJL-H2s C3c/1CyJ, DBA2/J, or SJL/J mice were immunized with recombinant murine Dsg3 (mDsg3), (ii) DBA2/J and SJL/J mice were immunized with mDsg3 and additionally injected a single non-blister inducing dose of exfoliative toxin A (ETA), and (iii) DBA2/J and SJL/J mice were immunized with human Dsg (hDsg) 1 and 3.

Results: Despite the induction of autoantibodies in each immunization protocol, the mice did not develop a clinical phenotype. Tissue-bound autoantibodies were not detected in the skin or mucosa. Circulating autoantibodies did not bind to the native antigen in indirect immunofluorescence microscopy using monkey esophagus as a substrate.

Conclusion: Immunization with PV autoantigens induced non-pathogenic Dsg1/3 antibodies, but did not cause skin/mucous membrane disease in mice. These findings, confirmed by failure of binding of the induced autoantibodies to their target in the skin, suggest that the autoantibodies which were formed were unable to bind to the conformational epitope present in vivo.

Publication types

  • Research Support, Non-U.S. Gov't

MeSH terms

  • Animals
  • Autoantibodies / immunology*
  • Desmoglein 3 / immunology*
  • Enzyme-Linked Immunosorbent Assay
  • Exfoliatins / therapeutic use
  • Female
  • Fluorescent Antibody Technique, Direct
  • Fluorescent Antibody Technique, Indirect
  • Male
  • Mice
  • Mice, Inbred C57BL
  • Pemphigus / immunology

Substances

  • Autoantibodies
  • Desmoglein 3
  • Exfoliatins

Grants and funding

This work was supported by the Research Training Group “Modulation of Autoimmunity” (RTG 1727/1-2) to DZ, ES and RJL, the Excellence Cluster “Precision Medicine in Chronic Inflammation” (EXC 2167) to DZ, ES and RJL, the individual grant LU 877/19-1 to RJL and by FOR 2497 Pemphigus - from Pathogenesis to Therapeutics (PEGASUS) to RE. All funding was provided by the Deutsche Forschungsgemeinschaft. The funders had no role in study design, data collection and analysis, decision to publish, or preparation of the manuscript.