BackgroundChoriocarcinoma coexisting with endometrial carcinoma is rare. To the best of our knowledge, only one case of choriocarcinoma coexisting with endometrial carcinoma has been reported.Case presentationHere, we present this case and provide a literature review. A 38-year-old unmarried nulliparous woman presented to the clinic with a menstrual disorder for more than 3 months. She then underwent a hysteroscopic procedure. The pathological findings were malignant, two types of carcinoma, and no transitional lesions were observed; about 85% of them were choriocarcinoma with smooth muscle infiltration and intravascular investigation of the thrombus; about 15% were highly differentiated endometrioid adenocarcinoma; Immunohistochemistry (endometrioid/choriocarcinoma): Vim (+ + / +  + +), P40 (+ ±), CK5/6 multifocal ( ±), CK7 ( ±), EMA (+ ±), P16 multifocal ( ±), P53 (+ / + +), WT-1 (-/ + +), hCG (-/ +  + +), CD138 (-/ +  + +), Gly-3 (-/-), ER ( ±), PR (+ ±), Sall-4 (-/-), P21 (-/ +), P27 (-/ +  + +), CyclinE (-/ + +), Ki67 positivity rate (10%/95%). We performed a laparoscopic hysterectomy, bilateral adnexectomy, and pelvic and para-abdominal lymph node dissection after five cycles of chemotherapy. She was diagnosed with choriocarcinoma with endometrial cancer, stage IVb choriocarcinoma and stage IA endometrial cancer. Postoperative radiochemotherapy was administered. The patient was disease-free 40 months after the treatment ended.ConclusionWe report a case of choriocarcinoma coexisting with endometrial carcinoma and provide a literature review that may help inspire additional studies in the future.