Objective: To report an extremely rare case of spontaneous uterine perforation of choriocarcinoma with negative beta-human chorionic gonadotropin (β-hCG) post-chemotherapy. Clinical Presentation and Intervention: We present a 35-year-old choriocarcinoma patient whose serial serum β-hCG levels following a fifth course of chemotherapy had been within the normal range, but who developed spontaneous uterine perforation with intra-abdominal hemorrhage after eight courses of combined chemotherapy. The patient then underwent an emergency hysterectomy and survived. Conclusion: Patients with persistent focus of disease in the uterus might experience uterine perforation even after adequate chemotherapy, and therefore, the follow-up for patients after chemotherapy is very important.

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