The management of late gestational trophoblastic disease recurrence is challenging. We present a case of a 16-year-old woman who was diagnosed with a gestational trophoblastic neoplasia 14 months after her hydatidiform mole pregnancy. A staging was performed revealing only an intramural lesion, which resembled a myoma, in the fundus of the uterus. Despite two course of methotrexate, the human chorionic gonadotropin (hCG) level increased slowly. The presentation was highly suggestive for a placental site trophoblastic tumor. A local resection of the tumor by hysterotomy was performed. Pathologic examination revealed a choriocarcinoma with tumor-free surgical margins. Subsequently, the patient received three cycles of EMA-CO (etoposide, methotrexate, actinomycin, cyclophosphamide, and vincristine). At present, 89 months after the hysterotomy, the patient is well, with no sings of recurrence. This report illustrates that it is mandatory to have a histologic diagnosis of chemoresistant gestational trophoblastic neoplasia before performing definitive surgery.