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Background: Persistent left superior vena cava is a rare but important congenital vascular anomaly. However, PLSVC with absent RSVC (isolated PLSVC) is a very rare venous malformation We report on a rare case of persistent left superior vena cava (PLSVC) with absent right superior vena cava (RSVC). Case Report: This venous malformation was identified incidentally in a 69-year-old woman during chest multi-detector computed tomography (MDCT). On chest MDCT, the SVC was noted on the left side. A bridging vein drained the right jugular and right subclavian veins and joined the left brachiocephalic vein in order to form the PLSVC, which descended on the left side of the mediastinum and drained into the left atrium (LA). The patient had no additional cardiac anomaly. Conclusions: Isolated PLSVC is usually asymptomatic but it can pose difficulties for establishing central venous access, pacemaker implantation and cardiothoracic surgery. This condition is also associated with an increased incidence of congenital heart disease, arrhythmias and conduction disturbances. A wide spectrum of clinicians should be aware of this anomaly, its variations and possible complications.