Pemphigoid gestationis with predominant involvement of oral mucous membranes and IgA autoantibodies targeting the C-terminus of BP180

J Am Acad Dermatol. 2002 Nov;47(5):780-4. doi: 10.1067/mjd.2002.113677.

Abstract

Pemphigoid gestationis (PG) is an autoimmune pregnancy-associated subepidermal blistering disease. It usually affects skin and, rarely, mucous membranes. In the vast majority of patients with PG, the autoimmune response is directed to the membrane-proximal NC16A domain of the 180-kd bullous pemphigoid (BP) antigen (BP180) and is mediated by IgG1 and IgG3 autoantibodies. We report the case of a patient with PG associated with extensive lesions on oral mucous membranes. Immunoblotting studies demonstrated the presence of circulating IgA autoantibodies in the patient's serum that were exclusively directed to a 49 amino acid stretch on the C-terminal portion of the BP180 ectodomain located 800 amino acids downstream from NC16A. This C-terminal stretch of BP180 has previously been demonstrated to localize to the lamina lucida/lamina densa interface and to be recognized by IgG and IgA antibodies in a subgroup of patients with cicatricial pemphigoid as well as by IgG autoantibodies in some BP sera. Our patient's lesions healed without scarring within 6 weeks after delivery of a healthy child. The findings in this patient extend the clinical and immunopathologic spectrum of PG.

Publication types

  • Research Support, Non-U.S. Gov't

MeSH terms

  • Adult
  • Antibodies, Anti-Idiotypic / analysis*
  • Autoantigens / immunology*
  • Collagen Type XVII
  • Female
  • Fluorescent Antibody Technique, Indirect
  • Humans
  • Immunoblotting
  • Immunoglobulin A / analysis
  • Immunoglobulin A / immunology*
  • Immunosorbent Techniques
  • Mouth Mucosa / immunology*
  • Non-Fibrillar Collagens
  • Pemphigoid Gestationis / immunology*
  • Pemphigoid, Bullous / immunology*
  • Pregnancy

Substances

  • Antibodies, Anti-Idiotypic
  • Autoantigens
  • Immunoglobulin A
  • Non-Fibrillar Collagens