ACE Inhibitor Reaction‐Induced Acute Angioedema Leading to Macroglossia: Two Case Studie

Abstract

Macroglossia, either congenital or acquired enlargement of the tongue, is a rare condition that leads to functional deficiencies in speech, mastication/deglutition, and airway patency. Acute‐acquired cases of macroglossia are potentially life threatening and have myriad etiologies. Presented here are two cases of macroglossia in adults caused by acute angioedema resulting from reaction to angiotensin‐converting enzyme (ACE) inhibitors. The mechanism of ACE inhibitor‐induced angioedema is attributed to dysfunction in the kallikrien‐kinin system, a protease pathway converting high molecular weight kininogens to kinins such as bradykinin, which can lead to inflammation of the cardiovascular system. This condition, while rare in adults, is more easily diagnosed, monitored, and treated if detected early. However, many clinicians fail to educate patients on the warning signs of acute angioedema: swelling of the tongue, submandibular and pharyngeal spaces, and/or difficulty breathing. Furthermore, patients often do not recognize the symptoms of ACE inhibitor reactions, leading to delays in diagnosis and treatment. Management of acute angioedema and subsequent macroglossia includes hospitalization and intubation for airway maintenance, partial glossectomy, and subsequent glossal rehabilitation and speech therapy. Both cases presented here led to anoxic brain injury and necessitated intensive surgical intervention, demonstrating the need for better patient‐clinician communication for positive outcomes of ACE inhibitor use. Current research on the kinin system and its role in the cardiovascular system is limited, so baseline clinician‐patient education represents the best first‐line defense against life‐threatening cases of acute angioedema.Support or Funding InformationThis research is supported in part by funding from the Texas A&M University College of Dentistry and the Baylor University Medical Center at Dallas.This abstract is from the Experimental Biology 2018 Meeting. There is no full text article associated with this abstract published in The FASEB Journal.