Angiotensin-Converting Enzyme Inhibitor-Associated Angioedema: From Bed to Bench

J Investig Allergol Clin Immunol. 2020;30(4):272-280. doi: 10.18176/jiaci.0458. Epub 2019 Oct 10.

Abstract

Background and objective: Angiotensin-converting enzyme inhibitor-associated angioedema (ACEI-AAE) affects 0.1%-0.7% of patients treated with ACEIs. While previous research suggests that angioedema attacks result from increased vascular permeability, the pathogenesis is not completely understood. Objective: This study aimed to describe the clinical, genetic, and laboratory parameters of ACEI-AAE patients and to investigate the role of vascular endothelial growth factors A and C (VEGF-A and VEGF-C), angiopoietins 1 and 2 (Ang1/Ang2), and secretory phospholipase A2 (sPLA2) in the pathogenesis of ACEI-AAE.

Methods: The clinical and laboratory data of ACEI-AAE patients were collected from 2 angioedema reference centers. Healthy volunteers and ACEI-treated patients without angioedema were enrolled to compare laboratory parameters. Genetic analyses to detect mutations in the genes SERPING1, ANGPT1, PLG, and F12 were performed in a subset of patients.

Results: A total of 51 patients (57% male) were diagnosed with ACEI-AAE. The average time to onset of symptoms from the start of ACEI therapy was 3 years (range, 30 days-20 years). The most commonly affected sites were the lips (74.5%), tongue (51.9%), and face (41.2%). Switching from ACEIs to sartans was not associated with an increased risk of angioedema in patients with a history of ACEIAAE. VEGF-A, VEGF-C, and sPLA2 plasma levels were higher in ACEI-AAE patients than in the controls. Ang1/2 concentrations remained unchanged. No mutations were detected in the genes analyzed.

Conclusions: Our data suggest that sartans are a safe therapeutic alternative in ACEI-AAE patients. Increased concentrations of VEGF-A, VEGF-C, and sPLA2 in ACEI-AAE patients suggest a possible role of these mediators in the pathogenesis of ACEI-AAE.

Keywords: Angioedema; Angiotensin-converting enzyme inhibitor; Biomarkers; C1-inhibitor; Genetic analysis; Phospholipases A2; Vascular endothelial growth factor A; Vascular endothelial growth factor C.

MeSH terms

  • Adult
  • Aged
  • Aged, 80 and over
  • Angioedema / immunology*
  • Angiopoietin-1 / blood
  • Angiopoietin-2 / blood
  • Angiotensin II Type 1 Receptor Blockers / therapeutic use
  • Angiotensin-Converting Enzyme Inhibitors / adverse effects*
  • Angiotensin-Converting Enzyme Inhibitors / therapeutic use
  • Antigens, Human Platelet / blood*
  • Female
  • Humans
  • Male
  • Middle Aged
  • Risk
  • Treatment Switching
  • Up-Regulation
  • Vascular Endothelial Growth Factor A / blood*
  • Vascular Endothelial Growth Factor C / blood*

Substances

  • ANGPT1 protein, human
  • ANGPT2 protein, human
  • Angiopoietin-1
  • Angiopoietin-2
  • Angiotensin II Type 1 Receptor Blockers
  • Angiotensin-Converting Enzyme Inhibitors
  • Antigens, Human Platelet
  • VEGFA protein, human
  • Vascular Endothelial Growth Factor A
  • Vascular Endothelial Growth Factor C
  • human platelet antigen 1b