Mucocutaneous manifestations of the hyper-IgM immunodeficiency syndrome

J Am Acad Dermatol. 1998 Feb;38(2 Pt 1):191-6. doi: 10.1016/s0190-9622(98)70239-7.

Abstract

Background: The recurrent pyogenic infections of patients with hyper-IgM syndrome are controlled by intravenous gamma globulin administration, but patients may suffer from early-onset oral ulcerations and warts.

Objective: We have characterized the mucocutaneous manifestations associated with this condition to allow physicians to more readily identify it.

Methods: Three male patients with the mucocutaneous manifestations of the hyper-IgM syndrome are described. In one, histopathologic examination of the oral mucosal lesion was performed.

Results: Recurrent large, painful oral ulcerations can occur that are not necessarily associated with neutropenia nor do they respond to granulocyte colony-stimulating factor administration. Histopathologic examination of an ulcer showed a heavy infiltrate of mixed inflammatory cells. Warts tend to be widespread and resistant to traditional therapy.

Conclusion: Physicians should consider this uncommon condition when examining a male patient with severe oral ulcers or recalcitrant widespread warts.

Publication types

  • Case Reports

MeSH terms

  • Adult
  • Child
  • Child, Preschool
  • Dysgammaglobulinemia / complications*
  • Dysgammaglobulinemia / genetics
  • Genetic Linkage
  • Humans
  • Hypergammaglobulinemia / complications*
  • Hypergammaglobulinemia / genetics
  • Immunoglobulin M / blood*
  • Infections / complications
  • Male
  • Oral Ulcer / complications*
  • Oral Ulcer / pathology
  • Skin Diseases / complications*
  • Skin Diseases / pathology
  • Syndrome
  • X Chromosome

Substances

  • Immunoglobulin M