Lack of increased prevalence of immunoregulatory disorders in hereditary angioedema due to C1-inhibitor deficiency

Clin Immunol. 2011 Oct;141(1):58-66. doi: 10.1016/j.clim.2011.05.004. Epub 2011 May 19.

Abstract

Hereditary angioedema due to deficiency of C1-INH (HAE-C1-INH) is associated with enhanced consumption of the early complement components, which may predispose for autoimmune disease. We assessed the prevalence of such disorders among HAE- C1-INH patients and their impact on the natural course of HAE-C1-INH. Clinical data and immunoserological parameters of 130 HAE-C1-INH and 174 non-C1-INH-deficient patients with angioedema were analyzed. In our study, the incidence of immunoregulatory disorders was 11.5% in the population of HAE-C1-INH patients and 5.2% among non-C1-INH-deficient controls with angioedema. Immunoserology screening revealed a greater prevalence of anticardiolipin IgM (p=0.0118) among HAE-C1-INH patients, than in those with non-C1-INH-deficient angioedema. We did not find higher prevalence of immunoregulatory disorders among our HAE-C1-INH patients. However, in patients with confirmed immunoregulatory disorders, the latter influenced both the severity of HAE-C1-INH and the effectiveness of its long-term management. Appropriate management of the immunoregulatory disease thus identified improves the symptoms of HAE-C1-INH.

Publication types

  • Case Reports

MeSH terms

  • Adult
  • Angioedema / blood
  • Angioedema / complications
  • Angioedema / immunology
  • Antibodies, Anticardiolipin / blood
  • Autoantibodies / blood
  • Autoimmune Diseases / blood
  • Autoimmune Diseases / etiology*
  • Autoimmune Diseases / immunology
  • Case-Control Studies
  • Causality
  • Celiac Disease / blood
  • Celiac Disease / complications
  • Celiac Disease / immunology
  • Complement C1 Inhibitor Protein / metabolism*
  • Female
  • Hereditary Angioedema Types I and II / blood
  • Hereditary Angioedema Types I and II / complications*
  • Hereditary Angioedema Types I and II / immunology*
  • Humans
  • IgA Deficiency / blood
  • IgA Deficiency / complications
  • IgA Deficiency / immunology
  • Immunoglobulin M / blood
  • Male
  • Middle Aged
  • Young Adult

Substances

  • Antibodies, Anticardiolipin
  • Autoantibodies
  • Complement C1 Inhibitor Protein
  • Immunoglobulin M