Kimihiro Nishino

Outcomes of cases with complete hydatidiform mole coexisting with a fetus: a single-center study

Abstract Background Complete hydatidiform moles coexisting with a fetus (CHMCF) are uncommon. Although CHMCF is associated with perinatal complications and post-molar gestational trophoblastic neoplasia (GTN), necessitating post-delivery chemotherapy, live birth remains feasible. This report presents 14 cases of CHMCF in Japan. Methods  We reviewed medical records of patients with CHMCF treated at our hospital from 2000 to 2020 and summarized clinical data, including maternal age, pregnancy details, delivery outcomes, fertility treatments, serum human chorionic gonadotropin (hCG) levels, and ultrasonography findings. Results Fourteen cases of CHMCF were diagnosed. The average age of the mothers was 30.6 years, with the majority conceiving following fertility treatment. The mean gestational age at diagnosis was 12 weeks. Six patients maintained their pregnancies, leading to two live births through emergency cesarean section. Eight patients exhibited spontaneous regression following treatment and pregnancy interruption, achieving negative serum hCG levels within 17.4 weeks. Six patients experienced post-molar GTN, including the two who had live births. One patient presented with FIGO stage I disease, while five patients had stage III lung metastases. All patients received chemotherapy, averaging nine courses, achieving remission within 13.7 weeks. Conclusion  The occurrence of GTN was higher after CHMCF than after typical complete hydatidiform moles. Despite the heightened risk of premature birth, some patients with CHMCF who maintain their pregnancies can successfully deliver live babies. Informed consent is essential for patients with CHMCF when considering pregnancy continuation. A team approach involving gynecological oncologists, obstetricians, and neonatologists is essential for effective diagnosis and treatment.

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 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.