Anomalous unilateral single pulmonary vein is a rare abnormality of the pulmonary venous system characterized by a tortuous pulmonary venous confluence which never crosses the diaphragm and drains all arterialized blood from a lung to the left atrium, into which it normally flows. Cardiac and pulmonary anomalies are not associated. We report a case in which this condition was comfirmed by the findings of chest radiography, computed tomography, and magnetic resonance angiography.