There is no doubt that appropriate use of thrombolytic drugs improves survival in patients with acute myocardial infarction. Streptokinase, one of the most commonly used thrombolytic drugs in large clinical trials, is not fibrin-specific and brings about a lytic state with potential for bleeding. We report a case of spontaneous rectus muscle hematoma following streptokinase therapy for acute myocardial infarction and review the relevant literature. To our knowledge, this is the first case reported in the literature.