Prognostic implications of determining 180 kDa bullous pemphigoid antigen (BPAG2) gene/protein pathology in neonatal junctional epidermolysis bullosa

Br J Dermatol. 1998 Apr;138(4):661-6. doi: 10.1046/j.1365-2133.1998.02182.x.

Abstract

Epidermolysis bullosa (EB) in neonates is often difficult to characterize, both in terms of making a precise diagnosis and in being able to comment accurately on the prognosis for the affected child. We present a case of a neonate with inherited mucocutaneous fragility and failure to thrive and detail our laboratory approach for classifying the subtype of EB in this child. Mutational analysis revealed a homozygous non-sense mutation in the gene encoding the 180 kDa bullous pemphigoid antigen, also known as type XVII collagen, predicting a non-lethal form of junctional EB. Identification of the underlying molecular pathology in this case was of use in improving diagnosis, classification, management and counselling.

Publication types

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

MeSH terms

  • Autoantigens / genetics*
  • Carrier Proteins*
  • Collagen Type XVII
  • Collagen*
  • Cytoskeletal Proteins*
  • Dystonin
  • Epidermolysis Bullosa, Junctional / diagnosis*
  • Epidermolysis Bullosa, Junctional / genetics
  • Epidermolysis Bullosa, Junctional / pathology
  • Female
  • Fluorescent Antibody Technique, Indirect
  • Humans
  • Infant, Newborn
  • Mutation
  • Nerve Tissue Proteins*
  • Non-Fibrillar Collagens*
  • Pedigree
  • Prognosis

Substances

  • Autoantigens
  • Carrier Proteins
  • Cytoskeletal Proteins
  • DST protein, human
  • Dystonin
  • Nerve Tissue Proteins
  • Non-Fibrillar Collagens
  • Collagen