This is the first report of ovarian cancer developing in a patient with HIV infection. The patient had Stage IIIC, grade 2, serous ovarian carcinoma. Aggressive therapy consisting of primary cytoreduction, high-dose platinum-based chemotherapy, and second-look laparotomy was well tolerated. The patient's CA-125 level quickly normalized but microscopic disease was found at second-look laparotomy. Although ovarian carcinoma has not been reported in the HIV-positive population, the incidence is likely to increase as the HIV-positive patient population ages and the epidemic in women continues. Aggressive primary therapy can result in a significant response; however, further experience and follow-up will be necessary to determine if there is decreased survival in this patient group as has been reported with other HIV-associated malignancies.