Clinical Pattern and Acute and Long-term Management of Hereditary Angioedema Due to C1-Esterase Inhibitor Deficiency

J Investig Allergol Clin Immunol. 2015;25(5):358-64.

Abstract

Background: Hereditary angioedema due to C1-esterase inhibitor deficiency (HAE-C1-INH) is a life-threatening disease.

Objectives: To describe the clinical characteristics and management of patients with HAE-C1-INH during routine clinical practice.

Methods: An observational, retrospective study was performed in patients with HAE-C1-INH. Demographic, clinical, and analytical data were collected from 2 periods: period A (October 2009-September 2010) and period B (October 2007-September 2009).

Results: We studied 112 patients with HAE-C1-INH (57.1% females). Age at onset of symptoms was 14.4 years (lower in patients who had experienced attacks in the previous year). In period B (n=87), 62.1% of patients presented at least 1 edema attack (median, 3.5 attacks/patient/2 years), and 19.1% of attacks were treated. In period A (n=77), 58.4% of patients were on maintenance therapy. Stanozolol was the most widely used drug (48.9%), with a mean weekly dose of 6.7 mg. At least 1 attack was recorded in 72.7% of patients (median, 3.0 attacks/patient/year), and 31.5% of the attacks were treated. Treatment of acute attacks increased by 12.4%.

Conclusion: Age at onset of symptoms is associated with clinical expression of disease. The higher age at onset of symptoms, the fewer number of attacks per patient and year, and the lower dose of attenuated androgens necessary to control the disease than in other series lead us to hypothesize that HAE-C1-INH could have a less severe expression in Spain. Acute attacks seem to be treated increasingly often.

Publication types

  • Observational Study
  • Research Support, Non-U.S. Gov't

MeSH terms

  • Adolescent
  • Adult
  • Aged
  • Androgens / therapeutic use*
  • Anti-Inflammatory Agents, Non-Steroidal / therapeutic use*
  • Antifibrinolytic Agents / therapeutic use*
  • Bradykinin / analogs & derivatives*
  • Bradykinin / therapeutic use
  • Child
  • Child, Preschool
  • Complement C1 Inhibitor Protein / therapeutic use*
  • Disease Management
  • Female
  • Hereditary Angioedema Types I and II / drug therapy*
  • Hereditary Angioedema Types I and II / etiology
  • Hereditary Angioedema Types I and II / physiopathology
  • Humans
  • Male
  • Middle Aged
  • Retrospective Studies
  • Young Adult

Substances

  • Androgens
  • Anti-Inflammatory Agents, Non-Steroidal
  • Antifibrinolytic Agents
  • Complement C1 Inhibitor Protein
  • icatibant
  • Bradykinin