Recurrence of unilateral angioedema of the tongue: A case report

BACKGROUND: Angioedema is a disorder characterized by edema of the face, lips, tongue, and extremities due to increased vascular permeability. Angioedema of the tongue usually occurs bilaterally, and the incidence of unilateral angioedema of the tongue is rare. This study reports a rare case of unilateral angioedema of the tongue with no identifiable cause and repeated recurrence even after discontinuation of an angiotensin-converting enzyme inhibitor. CASE SUMMARY: The patient was a 65-year-old woman with pre-existing hypertension and hyperlipidemia and had been receiving 20 mg/d of lisinopril. She was diagnosed with angioedema due to unilateral swelling of the tongue. No airway obstruction was observed, and the symptoms improved following the administration of 0.3 mg of epinephrine intramuscularly and 125 mg of methylprednisolone intravenously. Although lisinopril was discontinued, unilateral angioedema of the tongue continued to recur every 2-5 mo, with the symptoms improving following the administration of prednisolone and an antihistamine. Daily oral administration of 500 mg of tranexamic acid after dinner prevented the recurrence of angioedema. CONCLUSION: Careful monitoring and identification of the underlying mechanism play a crucial role in the treatment of angioedema.