Hypocomplementemic urticarial vasculitis syndrome is associated with high levels of serum IgG4: a clinical manifestation that mimics IgG4-related disease

Intern Med. 2011;50(10):1109-12. doi: 10.2169/internalmedicine.50.4515. Epub 2011 May 1.

Abstract

A 58-year-old Japanese woman presented with recurrent abdominal pain, chronic urticaria, and petechiae on her extremities, and hypocomplementemia, findings that were consistent with hypocomplementemic urticarial vasculitis syndrome (HUVS). A laboratory examination revealed that she had markedly elevated IgG levels (4,448 mg/dL; normal range, 870-1,700 mg/dL) with particularly high IgG4 levels (1,050 mg/dL; normal range, 48-105 mg/dL) and a high IgG4/total IgG ratio (0.22; normal range, 0.02-0.05). A skin biopsy demonstrated leukocytoclastic vasculitis with IgG4 deposition in the vascular lumen and vascular walls. A lymph node biopsy revealed reactive lymphoid hyperplasia with numerous IgG4-positive cells in the perifollicular area, but no sclerotic findings. A chromosomal analysis of an enlarged lymph node, without phytohemagglutinin (PHA) stimulation, demonstrated that one in every three analyzed cells had abnormalities, such as 44, XX, -13, add(15)(p11), -17, -17, and mar.

Publication types

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

MeSH terms

  • Chromosome Aberrations
  • Complement System Proteins / deficiency*
  • Female
  • Humans
  • Hypergammaglobulinemia / blood
  • Hypergammaglobulinemia / complications*
  • Hypergammaglobulinemia / immunology*
  • Immunoglobulin G / blood*
  • Middle Aged
  • Syndrome
  • Urticaria / blood
  • Urticaria / complications*
  • Urticaria / genetics
  • Urticaria / immunology*
  • Vasculitis / blood
  • Vasculitis / complications*
  • Vasculitis / genetics
  • Vasculitis / immunology*
  • Vasculitis, Leukocytoclastic, Cutaneous / blood
  • Vasculitis, Leukocytoclastic, Cutaneous / complications
  • Vasculitis, Leukocytoclastic, Cutaneous / genetics
  • Vasculitis, Leukocytoclastic, Cutaneous / immunology

Substances

  • Immunoglobulin G
  • Complement System Proteins