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Background: Angioedema, like anaphylaxis, has been reported as a rare adverse event of alteplase infusion in acute ischemic stroke. Objective: We report the case of a patient with acute ischemic stroke who, after treatment of alteplase, developed angioedema. Methods: We report the case of an 81-year-old woman who presented to our observation with acute ischemic stroke. The patient was on therapy with 100 mg acetylsalicylic acid and a triple combination antihypertensive drug (perindopril 10 mg + 2.5 mg indapamide + 5 mg amlodipine). The patient was treated with alteplase infusion. Results: Five minutes after the end of the alteplase infusion (0.9 mg/kg for 1 hour), the patient developed isolated angioedema of the lips and tongue. Conclusion: Although the incidence of alteplase-induced angioedema in these patients is rare, this case report suggests the need for a routine inspection of the tongue in an acute ischemic patient in treatment with alteplase infusion, especially in female patients in treatment with ACE inhibitors.