A child with localized vulval pemphigoid and IgG autoantibodies targeting the C-terminus of collagen XVII/BP180

Br J Dermatol. 1999 Jun;140(6):1133-8. doi: 10.1046/j.1365-2133.1999.02891.x.

Abstract

Localized vulval pemphigoid of childhood (LVPC) has previously been reported in six girls. Clinical features and immunopathological data have suggested it to be a morphological variant of bullous pemphigoid. Epitope targets of the autoantibodies of these patients have not been defined in detail. We describe a 9-year-old girl with possible cicatricial LVPC and circulating IgG antibodies directed against native collagen XVII/BP180, its 120-kDa soluble ectodomain and against the C-terminus of collagen XVII/BP180. No reactivity was detected towards the NC16A domain of collagen XVII/BP180. Linear IgG and C3 deposits were found along the cutaneous basement membrane zone. On 1 mol/L salt-split skin, IgG autoantibodies were shown to bind to the epidermis, and the HLA type II allele DQB1*0301, a marker with significantly increased occurrence in patients with ocular and oral cicatricial pemphigoid, was identified in this patient. Our data suggest that LVPC is a variant of bullous pemphigoid in which direct immunofluorescence microscopy combined with immunoblot analysis can deliver valuable diagnostic information for differential diagnosis. However, differentiation between the scarring and non-scarring course of the disease cannot be made with the present diagnostic markers and therefore careful follow-up of patients with LVPC is required.

Publication types

  • Case Reports
  • Research Support, Non-U.S. Gov't

MeSH terms

  • Autoantibodies / analysis*
  • Autoantibodies / blood
  • Biomarkers / analysis
  • Biomarkers / blood
  • Child
  • Collagen / immunology*
  • Complement C3 / analysis
  • Diagnosis, Differential
  • Electrophoresis, Polyacrylamide Gel
  • Epitope Mapping
  • Epitopes / immunology
  • Female
  • HLA-DQ Antigens / analysis
  • HLA-DQ beta-Chains
  • Humans
  • Immunoblotting
  • Immunoglobulin G / analysis*
  • Microscopy, Fluorescence
  • Pemphigus / diagnosis*
  • Skin / immunology*
  • Vulvar Diseases / diagnosis*

Substances

  • Autoantibodies
  • Biomarkers
  • Complement C3
  • Epitopes
  • HLA-DQ Antigens
  • HLA-DQ beta-Chains
  • HLA-DQB1 antigen
  • Immunoglobulin G
  • Collagen