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Metastatic and disseminated uterine leiomyosarcoma has limited chemotherapeutic options.A 45-year-old nulligravida was originally diagnosed with a primary uterine leiomyosarcoma and treated with a total abdominal hysterectomy, followed by six courses of adjuvant platinum and doxorubicin chemotherapy. Three years later she developed multiple metastatic tumor nodules in the lungs. Because of the estrogen receptor positivity of the leiomyosarcoma by immunohistochemistry, she was treated with 1 mg of anastrozole daily. Progressive tumor regression was demonstrated by serial imaging in all metastatic lung tumor deposits, with an objective response lasting at least 12 months.Uterine leiomyosarcoma metastatic to the lungs regressed with the use of the anastrozole aromatase inhibitor.