Only a small number of venous leiomyosarcomas have been previously reported. Of these tumors, those of saphenous origin comprise a minority of cases. A 59-year-old man presented with symptoms of deep vein thrombosis and was eventually diagnosed with primary leiomyosarcoma of great saphenous vein origin. The tumor was treated with primary resection and femoral vein reconstruction with autologous patch. Although extremely rare, saphenous leiomyosarcoma can present as deep vein thrombosis. Vascular tumors should be included in the differential diagnosis of atypical extremity swelling refractory to conventional deep vein thrombosis management.
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