Abstract
BackgroundPure ovarian choriocarcinoma can be gestational or nongestational in origin. Nongestational pure ovarian choriocarcinoma is extremely rare and the prognosis is thought to be worse than that of the gestational type in patients with metastatic disease. We present a case of metastatic pure ovarian choriocarcinoma with poor prognosis in which the origin was identified as nongestational by DNA short tandem repeat (STR) analysis.Case presentationA nulliparous woman in her thirties with metastatic choriocarcinoma was referred to our hospital after initial treatment proved unsuccessful. Two months earlier, she had undergone brain tumor resection and histological examination confirmed choriocarcinoma. Serum human chorionic gonadotropin (hCG) concentration at initial diagnosis was 5030 IU/L. Two cycles of a combination chemotherapy regimen of methotrexate, etoposide, and actinomycin-D (MEA therapy), which is commonly used for gestational choriocarcinoma, was administered. However, the disease could not be controlled. Imaging modalities at presentation revealed tumor present in the left ovary and left lung, but not in the uterus, which led us think that the choriocarcinoma was nongestational. Bleomycin, etoposide, and cisplatin (BEP therapy) which is commonly used for nongestational choriocarcinoma (malignant germ cell tumor) and surgical resection of the uterus, bilateral ovaries, and an affected part of the left lung led to the nadir level of hCG, but the tumor relapsed and levels of hCG again increased. To investigate the origin of choriocarcinoma, we performed DNA STR analysis of tumor cells and oral mucosal cells. Analysis revealed the origin of the choriocarcinoma as nongestational, as the genotype of tumor cells entirely corresponded with that of oral mucosal cells. BEP therapy and chemotherapy regimens administered for nongestational choriocarcinoma and gestational choriocarcinoma proved ineffective, and the patient died 21 months after diagnosis of metastatic choriocarcinoma.ConclusionMetastaic nongestational pure choriocarcinoma of ovary is an extremely rare and an aggressive disease, frequently resulting in poor outcome.
Highlights
Pure ovarian choriocarcinoma can be gestational or nongestational in origin
We report a case of metastatic primary pure ovarian choriocarcinoma identified as nongestational by DNA short tandem repeat (STR) analysis that showed chemo-resistance to regimens administered for gestational choriocarcinoma as well as nongestational choriocarcinoma with a review of the literature
Chest Computed tomography (CT) revealed metastatic choriocarcinoma in the left lung (Fig. 1c). Her disease was diagnosed as primary pure ovarian choriocarcinoma and two cycles of a combination chemotherapy regimen comprising methotrexate, etoposide, and actinomycin-D (MEA therapy) which is commonly used for gestational choriocarcinoma, were administered
Summary
Pure ovarian choriocarcinoma can be classified into two groups based on origin: gestational or nongestational. Chest CT revealed metastatic choriocarcinoma in the left lung (Fig. 1c) Her disease was diagnosed as primary pure ovarian choriocarcinoma and two cycles of a combination chemotherapy regimen comprising methotrexate, etoposide, and actinomycin-D (MEA therapy) which is commonly used for gestational choriocarcinoma, were administered. Two additional cycles of BEP therapy failed to achieve complete remission, and hCG level increased steeply when chest CT revealed multiple metastases in the lungs. Her clinical history was reevaluated to identify the origin of choriocarcinoma and choose an appropriate chemotherapy regimen. A combination chemotherapy regimen of MEA, paclitaxel and cisplatin (TP), fluorouracil and actinomycin-D (FA), ifosfamide, cisplatin, and etoposide (ICE), etoposide, methotrexate, actinomycin-D/cyclophosphamide and vincristine (EMA/CO) with segmentectomy of the right lung were administered after tumor relapse, but no
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