Orolingual angioedema (OLAE) is a rare adverse effect of alteplase. Previous studies have associated the occurrence of OLAE with thrombolysed patients maintained on angiotensin converting enzyme inhibitors. We report a case of a 60-year-old male presenting with hyperacute ischemic stroke developing hemilingual edema after thrombolysis. He was previously maintained on an angiotensin II receptor blocker (ARB), losartan. The swelling resolved over 2 days with immediate administration of intravenous steroids and antihistamine drugs.

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