We describe a patient who had recurrent life-threatening episodes of isolated macroglossia due to the use of an angiotensin-converting enzyme (ACE) inhibitor. No associated facial, labial, pharyngeal, or laryngeal edema was noted. Aggressive treatment with epinephrine, steroids, and antihistamines resulted in rapid resolution of the tongue swelling and respiratory distress. Recurrent isolated angioedema of the tongue is an extremely rare variant of ACE inhibitor-related angioneurotic edema. The widespread use of ACE inhibitors mandates a special awareness by physicians of this potentially life-threatening yet treatable side effect.
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KyrmizakisDEPapadakisCEFountoulakisEJLioliosADSkoulasJG. Tongue angioedema after long-term use of ACE inhibitors. Am J Otolaryngol1998; 19: 394–6
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DeanDESchultzDLPowersRH. Asphyxia due to angiotensin converting enzyme (ACE) inhibitor mediated angioedema of the tongue during the treatment of hypertensive heart disease. J Forensic Sci2001; 46: 1239–43
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GadbanHGilbeyPTalmonYSametA. Acute edema of the tongue: a life-threatening condition. Ann Otol Rhinol Laryngol2003; 112: 651–3
