Major complications of thrombolysis are intracranial and extracranial bleedings. Compartment syndrome (CS) as a serious adverse event is sparsely reported. The purpose of the study is to present a systematic review of the literature on this complication based on a case vignette. A PubMed and Google Scholar search on CS following thrombolysis was performed. Twenty-four patients (11 male, 11 female, 2 not noted; median age 66 years, range 19-85 years) with thrombolysis associated CS were identified. Fifteen patients had thrombolysis with rtPA, 4 patients with streptokinase, 3 patients with urokinase, and 2 patients with tenecteplase. In 15 cases, CS affected the upper limb, and in 9 cases the lower limb. Indication for thrombolysis was myocardial infarction in 11 patients, arterial occlusion of the leg in 6 patients, pulmonary embolism in 4 patients, stroke in 2 patients, and deep venous thrombosis in 1 patient. In addition, in 15 cases, aspirin/ticlopidin, and/or heparin in therapeutic dosages had been prescribed. In 15 cases manipulations of the affected limb had been preceding. In both stroke patients, a hidden fracture was later diagnosed. The median time to the diagnosis of CS was 12 h (2 h-3 days). Therapy was mostly surgical with fasciotomy. The outcome of CS was favorable in 14 cases. However, in 5 cases, nerve damage persisted, and amputation was indicated in 2 patients. CS following thrombolysis is a rare condition. As predisposing factors different manipulations, hidden fracture and pronounced antithrombotic therapy are encountered.