Uterine tumor resembling ovarian sex-cord tumor (UTROSCT) is a rare gynecological tumor which has undetermined pathogenesis but with distinct polyphenotypic immuno-histochemical expressions. According to the limited cases and follow-up information in the relevant literatures, most of the tumors exhibit indolent or low malignant clinical course and the outcomes of the patients with the tumors generally have a good ending. But for the subset of UTROSCT with aggressive characters, the outcomes of the patients with recurrent neoplasm were not always satisfactory. This case report reported neoplasm recurrence in the pelvic cavity after 53 months of surgery and irregular follow-up. The recurrent neoplasm grew in an invasive manner. The arrangement of the recurrent neoplasm cells was closer, the nucleus atypia was more pronounced, and the cells demonstrated a more briskly mitotic activity (10 mitotic figures per ten high-powered fields, 10 mitotic figures/10 HPF). The Ki67 index increased significantly. Both the clinical characteristics and histological morphology of the recurrent neoplastic cells showed a more malignant behavior. The patient received a palliative resection of pelvic mass and bilateral oophorectomy, and she died of intestinal obstruction caused by the recurrent disease 9 months postoperatively. UTROSCT with characters for the aggressive process should deserve more attention. Once it relapses, the recurrent tumor of UTROSCT might show a higher malignant tendency and a poor prognosis. Gynecologists and pathologists need to analyze the clinical and histologic assessment in collaboration and better evaluate UTROSCT with aggressive characters.