A poor prognostic metastatic nongestational choriocarcinoma of the ovary: a case report and the literature review
Abstract Background Pure 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 metastat...
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| Format: | Article |
| Language: | English |
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BMC
2021-04-01
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| Series: | Journal of Ovarian Research |
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| Online Access: | https://doi.org/10.1186/s13048-021-00810-3 |
| _version_ | 1828080823747739648 |
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| author | Kimihiro Nishino Eiko Yamamoto Yoshiki Ikeda Kaoru Niimi Toshimichi Yamamoto Hiroaki Kajiyama |
| author_facet | Kimihiro Nishino Eiko Yamamoto Yoshiki Ikeda Kaoru Niimi Toshimichi Yamamoto Hiroaki Kajiyama |
| author_sort | Kimihiro Nishino |
| collection | DOAJ |
| description | Abstract Background Pure 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 presentation A 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. Conclusion Metastaic nongestational pure choriocarcinoma of ovary is an extremely rare and an aggressive disease, frequently resulting in poor outcome. |
| first_indexed | 2024-04-11T03:25:21Z |
| format | Article |
| id | doaj.art-fbbaa2c23a30448aae39294c8366fe57 |
| institution | Directory Open Access Journal |
| issn | 1757-2215 |
| language | English |
| last_indexed | 2024-04-11T03:25:21Z |
| publishDate | 2021-04-01 |
| publisher | BMC |
| record_format | Article |
| series | Journal of Ovarian Research |
| spelling | doaj.art-fbbaa2c23a30448aae39294c8366fe572023-01-02T07:50:46ZengBMCJournal of Ovarian Research1757-22152021-04-011411610.1186/s13048-021-00810-3A poor prognostic metastatic nongestational choriocarcinoma of the ovary: a case report and the literature reviewKimihiro Nishino0Eiko Yamamoto1Yoshiki Ikeda2Kaoru Niimi3Toshimichi Yamamoto4Hiroaki Kajiyama5Department of Obstetrics and Gynecology, Nagoya University Graduate School of MedicineDepartment of Obstetrics and Gynecology, Nagoya University Graduate School of MedicineDepartment of Obstetrics and Gynecology, Nagoya University Graduate School of MedicineDepartment of Obstetrics and Gynecology, Nagoya University Graduate School of MedicineDepartment of Legal Medicine and Bioethics, Nagoya University Graduate School of MedicineDepartment of Obstetrics and Gynecology, Nagoya University Graduate School of MedicineAbstract Background Pure 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 presentation A 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. Conclusion Metastaic nongestational pure choriocarcinoma of ovary is an extremely rare and an aggressive disease, frequently resulting in poor outcome.https://doi.org/10.1186/s13048-021-00810-3Pure ovarian nongestational choriocarcinomaShort tandem repeat analysisBEP therapy |
| spellingShingle | Kimihiro Nishino Eiko Yamamoto Yoshiki Ikeda Kaoru Niimi Toshimichi Yamamoto Hiroaki Kajiyama A poor prognostic metastatic nongestational choriocarcinoma of the ovary: a case report and the literature review Journal of Ovarian Research Pure ovarian nongestational choriocarcinoma Short tandem repeat analysis BEP therapy |
| title | A poor prognostic metastatic nongestational choriocarcinoma of the ovary: a case report and the literature review |
| title_full | A poor prognostic metastatic nongestational choriocarcinoma of the ovary: a case report and the literature review |
| title_fullStr | A poor prognostic metastatic nongestational choriocarcinoma of the ovary: a case report and the literature review |
| title_full_unstemmed | A poor prognostic metastatic nongestational choriocarcinoma of the ovary: a case report and the literature review |
| title_short | A poor prognostic metastatic nongestational choriocarcinoma of the ovary: a case report and the literature review |
| title_sort | poor prognostic metastatic nongestational choriocarcinoma of the ovary a case report and the literature review |
| topic | Pure ovarian nongestational choriocarcinoma Short tandem repeat analysis BEP therapy |
| url | https://doi.org/10.1186/s13048-021-00810-3 |
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