AbstractBackground and purposeOrolingual angioedema (OA) represents a rare but life‐threatening complication among patients with acute ischemic stroke treated with intravenous thrombolysis with alteplase. Novel agents, including icatibant, are recommended in resistant patients with alteplase‐induced OA who have failed to respond to first‐line therapies including corticosteroids, antihistamines, and/or adrenaline.MethodsWe present a patient with alteplase‐induced OA who showed substantial clinical improvement following the administration of icatibant.ResultsWe describe a 71‐year‐old woman with known arterial hypertension under treatment with angiotensin‐converting enzyme inhibitor, who presented with acute ischemic stroke in the territory of the right middle cerebral artery and received intravenous alteplase. During intravenous thrombolysis, the case was complicated with OA without any response to standard anaphylactic treatment including corticosteroids, dimetindene, and adrenaline. Thirty minutes after symptom onset, icatibant, a synthetic selective bradykinin B2‐receptor antagonist, was administered subcutaneously. Substantial symptomatic resolution was observed only following the icatibant administration.ConclusionsThis case highlights the effectiveness of icatibant in the acute management of alteplase‐induced OA. In particular, icatibant administration, following first‐line therapies including corticosteroids, antihistamines, and/or adrenaline, may avert tracheostomy and intubation in resistant and refractory cases with OA following intravenous thrombolysis for acute ischemic stroke.