Nongestational ovarian choriocarcinoma (NGOC) is a very rare germ cell tumor that normally appears alongside other malignant germ cell tumors. We present a case of NGOC that occurred in the absence of any other malignant germ cell tumor. A 17-year-old virgin girl was referred to our hospital after experiencing atypical genital bleeding for two months. Magnetic resonance imaging revealed a 140×90 mm multilocular cystic mass with a partly solid portion in the left adnexal region, as well as a cystic mass, 30 mm in diameter, in the right adnexal region. Laparotomy was performed after the preliminary diagnosis of a bilateral mature cystic teratoma of the ovaries, and included a left salpingo-oophorectomy, right cystectomy, and resection of a mass in the pouch of Douglas. Histological examination of the tumor confirmed an ovarian choriocarcinoma, with no other malignant germ cell tumor, coexisting with a mature cyctic teratoma. The mass in the pouch of Douglas was a metastatic tumor arising from the choriocarcinoma. After surgery, the patient’s serum human chorionic gonadotropin (hCG) level showed 1211 mIU/mL and computed tomography (CT) revealed multiple lung metastases. The final postoperative diagnosis was made as NGOC, stage IV. A combination chemotherapy regimen of methotrexate, etoposide, and actinomycin-D (MEA) was administered five times. After the second course, serum hCG level dropped to within normal range. CT after completion of chemotherapy showed that the lung metastases had disappeared. Tumor recurrence in the uterus and multiple lung metastases were detected one year later. Four cycles of chemotherapy with bleomycin, etoposide, and cisplatin (BEP) were administered, but were ineffective. Thoracoscopic partial lung resection for the lung metastases, as well as total abdominal hysterectomy, were performed. Afterwards, adjuvant chemotherapy regimen was administered; however, the patient died 43 months after her initial presentation because of diffuse lung metastases. [Adv Obstet Gynecol, 68 (2) : 118-125, 2016 (H28.5)]
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