Only five patients found to have brain metastasis preceding the diagnosis of endometrial cancer have been reported in the literature, and none of these survived beyond 38 months. The authors report on two patients with primary endometrial cancer who initially presented with cerebral metastasis. One of these patients died of disease 15 months after diagnosis. The other patient is still alive, with no evidence of disease, 171 months after she underwent radiosurgery for a solitary brain metastasis, aggressive cytoreductive abdominal and pelvic surgery, and doxorubicin-based chemotherapy. To the best of their knowledge, the authors believe that no similar observation has been made for any primary gynecological neoplasm, including endometrial, ovarian, or cervical cancer. This is the first report documenting that survival beyond one decade may be achieved after intensive multimodal therapy in selected patients in whom a solitary brain metastasis has been found before diagnosis of endometrial cancer. Aggressive therapy appears to be warranted in these patients.