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The aim of this study was to evaluate the rate, clinical characteristics, and survival outcomes of an undiagnosed uterine malignancy in patients who underwent surgical treatment for presumed leiomyomas.Medical records of patients who underwent surgical treatment for presumed leiomyomas, from January 2004 to September 2013, were retrospectively reviewed, and the data were followed until September 2016. Demographic data, tumor characteristics, oncologic treatment, and response rate were analyzed by descriptive statistics. Kaplan-Meier method was used for survival analysis. This study includes follow-up data through December 31, 2016.A total of 11,258 medical records of presumed leiomyoma patients undergoing hysterectomy during the studied period were reviewed. Pathology of uterine sarcoma was found in 22 patients (0.2%), all of whom were included. Nineteen patients had leiomyosarcoma, and 3 had endometrial stromal sarcoma. Mean age of patients was 48.3 ± 6.9 years. All patients underwent total abdominal hysterectomy, with 20 patients undergoing concurrent bilateral salpingo-oophorectomy. Uterine sarcoma was classified as stage IB in 21 patients and stage IIIC in 1 patient. Fifteen patients were prescribed the following adjuvant treatment: chemotherapy in 13 patients and megestrol acetate in 2 patients. Thirteen patients had recurrent disease, and 3 patients died of their disease. The mean progression-free survival was 50.1 ± 41.3 months, and overall survival was 59.3 ± 43.0 months.One in 512 patients who underwent hysterectomy because of presumed uterine leiomyomas had inadvertent uterine sarcomas. Even with adjuvant therapy, treatment outcome was rather poor, with almost 60% recurrence rate and median progression-free survival and overall survival of less than 5 years.