A Rare Case of Unilateral Tongue Edema with Angiotensin Converting Enzyme Inhibitors

Abstract

Angiotensin converting enzyme inhibitors (ACEi) are widely used for the treatment of multiple conditions such as hypertension, heart failure and chronic kidney disease. Angioedema is a rare but potentially fatal complication of ACEi use and unilateral tongue edema is a very rare presentation. We report a case of a 55-year-old man, with a history of hypertension, on enalapril for three years, who presented to the hospital with unilateral tongue swelling, without airway compromise. Other causes were excluded and the diagnosis of angioedema due to enalapril was established. The patient was discharged with discontinuation of ACEi with total resolution of symptoms and without relapse after several months. Although very rare, unilateral tongue swelling should be considered in the presentation of angioedema associated with ACEi. Tight surveillance is important to prevent fatal complications such as airway obstruction. ACEi discontinuation is crucial to avoid clinical relapse.

Recurrence of unilateral angioedema of the tongue: A case report

“…A search of the database of PubMed using the keyword “unilateral tongue angioedema” retrieved 21 articles and 17 case reports (last retrieved on August 23, 2023). Among the retrieved results, details of the 15 cases that were reported in English are presented in Table 3 [ 3 - 16 ]. The median age of the patients was 68 years (range: 30-80 years).…”

“…Angioedema of the tongue usually occurs bilaterally, and cases of unilateral angioedema of the tongue are rare. The most frequently reported cause of unilateral tongue angioedema is drugs, such as ACE inhibitors and recombinant tissue-type plasminogen activators (rtPA), and recurrence of angioedema has not been reported after discontinuation of these drugs[ 3 - 16 ]. Herein, we describe a rare case of unilateral angioedema of the tongue with no identifiable cause that recurred repeatedly even after discontinuation of an ACE inhibitor.…”