Uterine adenomyosis is a common benign pelvic tumor in women. One of the complications that may be infrequently associated with the huge pelvic mass is venous stasis of the lower extremities and may develop thrombophlebitis secondary to pelvic compression. Intravascular thrombosis in the deep vein of the legs is a serious illness that sometimes cause death due to acute pulmonary thromboembolism. Deep vein thrombosis (DVT) has been reported to be closely related to pregnancy, surgical procedure, long term bedrest, obesity, and oral contraceptives. However, there is few report about deep vein thrombosis caused by huge uterine adenomyosis. We report a case of deep vein thrombosis caused by huge adenomyosis was treated by hysterectomy, thrombectomy, and thrombolysis with a brief review of the literature.