Thrombosis is an important complication of central venous catheterization. Among the many intrinsic and extrinsic factors, the patient's medical disease can play a role in thrombogenesis. Behcet's disease (BD), classified as a vasculitis, is a multisystem disease involving the small blood vessels. It is often difficult to recognize and diagnose the disease. A 24-yr-old female patient showed massive central venous thrombosis which caused superior vena cava syndrome after subclavian vein catheterization. Twenty days after catheterization, the patient exhibited swelling of the face, neck, and both upper extremities. Despite thrombectomy and continuous anticoagulation therapy, her facial and upper extremity swelling reappeared and follow-up chest computed tomography (CT) showed the recurrent thrombosis in the same central veins previously affected. A diagnosis of BD was then made. Following steroid therapy, neither clinical symptoms nor CT findings suggestive of central venous thrombosis were observed during the subsequent 6-months of follow-up period. This case emphasizes that central venous catheterization in a patient with BD should be performed with great caution.