IgA pemphigus--occurrence of anti-desmocollin 1 and anti-desmoglein 1 antibody reactivity in an individual patient

J Dtsch Dermatol Ges. 2006 Dec;4(12):1045-50. doi: 10.1111/j.1610-0387.2006.06166.x.
[Article in English, German]

Abstract

Background: IgA pemphigus is a rare pustular autoimmune disease with exclusive IgA anti-keratinocyte cell surface antibody reactivity. Two subtypes have been discerned: in the subcorneal pustular dermatosis type, desmocollin 1 has been identified as a targeted autoantigen, while in few cases of the intraepidermal neutrophilic type, IgA anti-desmoglein 1 or IgA anti-desmoglein 3 reactivity has been demonstrated.

Patients and methods: A 48-year-old white male presented with generalized large confluent pustules. Skin pathology was assessed by histology and direct immunofluorescence analysis. IgG/IgA autoantibodies against desmoglein 1/3 and desmocollin 1 were measured by ELISA and indirect immunofluorescence using desmocollin 1 cDNA-transfected COS7 cells, respectively.

Results: Histopathology revealed subcorneal pustules and direct immunofluorescence microscopy exclusively showed in vivo bound IgA with an intercellular pattern in the epidermis. Desmocollin 1 was identified as a target of IgA autoantibodies by indirect immunofluorescence microscopy utilizing desmocollin 1 cDNA-transfected COS7 cells. In addition, IgA anti-desmoglein 1 reactivity was demonstrated by ELISA. Neither IgA anti-desmoglein 3 nor IgG anti-desmoglein 1/3 autoantibodies were present.

Conclusions: Both desmocollin 1 and desmoglein 1 were autoantigens in this patient with IgA pemphigus and a distinct clinical presentation. To our knowledge, this is the first IgA pemphigus case with dual autoantibody reactivity.

Publication types

  • Case Reports
  • Comparative Study

MeSH terms

  • Anti-Bacterial Agents / administration & dosage
  • Anti-Bacterial Agents / therapeutic use
  • Anti-Infective Agents / administration & dosage
  • Anti-Infective Agents / therapeutic use
  • Anti-Inflammatory Agents / administration & dosage
  • Anti-Inflammatory Agents / therapeutic use
  • Autoantibodies / analysis
  • Autoantibodies / immunology
  • Autoantigens / immunology
  • Cefamandole / administration & dosage
  • Cefamandole / therapeutic use
  • Dapsone / administration & dosage
  • Dapsone / therapeutic use
  • Desmocollins / immunology*
  • Desmoglein 1 / immunology*
  • Enzyme-Linked Immunosorbent Assay
  • Fluorescent Antibody Technique
  • Humans
  • Immunoglobulin A* / immunology
  • Male
  • Middle Aged
  • Ointments
  • Pemphigus / drug therapy
  • Pemphigus / immunology*
  • Pemphigus / pathology
  • Skin / pathology
  • Sulfadiazine / administration & dosage
  • Sulfadiazine / therapeutic use
  • Time Factors
  • Treatment Outcome

Substances

  • Anti-Bacterial Agents
  • Anti-Infective Agents
  • Anti-Inflammatory Agents
  • Autoantibodies
  • Autoantigens
  • Desmocollins
  • Desmoglein 1
  • Immunoglobulin A
  • Ointments
  • Sulfadiazine
  • Cefamandole
  • Dapsone