Infective endocarditis represents a classical contra-indication to thrombolysis for acute ischemic stroke due to a potential increased risk of intracranial hemorrhage. However, some case reports have suggested safety and potential efficacy of intravenous or intra-arterial thrombolysis in stroke related to infective endocarditis. We present a case of ischemic stroke related to infective endocarditis who was treated with intravenous tissue plasminogen activator within the first 3 h of symptoms onset and subsequently developed symptomatic multifocal intracerebral hemorrhages, and summarize currently available data on this issue.