Central nervous system metastases are an unusual sequela of uterine sarcomas. A single brain metastasis from a leiomyosarcoma occurred in a 60-year-old woman, 6.5 years after hysterectomy and more than 2 years following the diagnosis of bilateral lung metastases, treated by combination chemotherapy. After resection of the cerebral metastasis, she was treated with brain radiation therapy and survived 2.5 years. Only seven other cases of uterus leiomyosarcoma metastatic to the brain have been published.