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Benign Metastasizing Leiomyoma (BML) is a rare disease which represents with multiple leiomyomatous lesions in many tissues and organs especially in lungs. These patients have been operated for leiomyoma of the uterus. Here we report a case of a 41-year-old woman who was evaluated in a thoracic surgery hospital for dyspnea and bilateral nodules in chest roentgenogram. She had no history of neoplasm, only myomectomy history of uterine leiomyoma 10 years ago. Biopsy and histopathological examination were consistent with pulmonary leiomyoma. The patient was reffered to our clinic and we performed a total abdominal hysterectomy for her multiple uterine leiomyomas. The final diagnosis was 'benign pulmonary metastasizing leiomyoma'. After this diagnosis, surgical castration was performed but two years later, repeat imaging showed progression in pulmonary lesions and progesterone therapy was administered to the patient. Patient has continued on this hormonal therapy to date and during the 5-years follow-up, the persisting lesions in both lungs regressed. [ABSTRACT FROM AUTHOR]