Leiomyosarcoma arising from the ovarian vein is extremely rare; we present a case with this unusual finding. A 78-year-old woman, diagnosed 2 years prior with a left retroperitoneal mass located in the lower part of the left kidney, was admitted to our hospital with a decrease in oral intake and a palpable, hard, abdominal mass. Contrast-enhanced CT showed a solid mass in the left lower abdominal cavity. On three-dimensional (3D) angiography the mass appeared to originate from the left ovarian vein. A simple total excision, including the involved vein, was performed and the tumour was found to be leiomyosarcoma. The patient's postoperative course was uneventful. There was no evidence of recurrence 5 months after surgery. In this patient, the previous information about the location of the retroperitoneal mass as well as the 3D angiography results were helpful in giving preoperative evidence of leiomyosarcoma originating from the left ovarian vein.