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Backgound: Endometrial mixed carcinoma with the neuroendocrine carcinoma (NEC) component is rare and is believed to have a poor prognosis. CD10 expression is reported to be a favorable prognostic marker for some tumors such as B-lymphoblastic leukemia/lymphoma, but unfavorable for others. Here, we report the case of a 33-year-old woman diagnosed with endometrial mixed carcinoma with the NEC component expressing CD10 who showed a favorable outcome., Case Presentation: The patient presented with lumbago and brownish discharge from the genitals. Imaging modalities revealed a large exophytic mass in the uterine corpus, and a small one in the uterine cervix. Radical hysterectomy with bilateral salpingo-oophorectomy was performed. Microscopic examination of the endometrial and cervical masses revealed that the NEC component accounted for the maximum area in both masses. However, small areas in both lesions showed well differentiated endometrioid adenocarcinoma (WDEA) components, and histological transition between the two components was also observed. In addition to CD56 and synaptophysin expression, the NEC component was positive for CD10 but negative for estrogen receptor (ER), progesterone receptor (PgR), and carcinoembryonic antigen (CEA). In contrast, the WDEA component expressed both ER and PgR, but neither CD10 nor neuroendocrine markers were demonstrated. The CD10 and neuroendocrine markers clearly distinguished between the NEC and WDEA components. Furthermore, retained expression of phosphatase and tensin homolog (PTEN) and weak phosphorylated Akt expression were found, which were assumed to suppress the aggressive behavior of the tumor. The patient received postoperative chemotherapy and has survived without recurrence for 6 years after the operation., Conclusion: This is the first case of endometrial mixed carcinoma with the NEC component expressing CD10 that showed a long survival.