Multiple metastatic gestational trophoblastic disease after a twin pregnancy with complete hydatidiform mole and coexisting fetus, following assisted reproductive technology: Case report and literature review

Objective: Twin pregnancy with complete hydatidiform mole and coexisting fetus (CHMCF) is rare and associated with severe complications during pregnancy and subsequent gestational trophoblastic disease (GTD). We encountered a case of multiple metastatic GTD after a twin pregnancy with CHMCF, following conventional in vitro fertilization (IVF). Only one case of metastatic GTD after CHMCF due to assisted reproductive technology (ART) has been reported. Here, we present the clinical course and reveal the clinical features of CHMCF after ART through a literature review. Case report: A 42-year-old primigravida woman had an abnormal pregnancy (i.e., CHMCF) by IVF. She had persisting severe vaginal bleeding, which led to termination of her pregnancy at 10 weeks of gestation. Pathohistological examination revealed that this was a case of CHMCF. Five weeks after the termination, the serum β-human chorionic gonadotropin level was still extremely high, and systemic contrast-enhanced computed tomography revealed a tumor in the uterine corpus and more than 30 lung nodules. After 11 cycles of combination chemotherapy with etoposide, methotrexate, actinomycin-D, cyclophosphamide, and vincristine (EMA/CO) to treat high-risk GTD, hysterectomy was needed as radical therapy. Conclusion: Cases of CHMCF following ART may also have higher malignant potential and higher risk of GTD development and become more aggressive biologically. The clinical course of CHMCF after ART seems to be almost the same as that without ART based on the results of literature review. Keywords: Assisted reproductive technologies, Complete hydatidiform mole coexisting with a fetus, EMA/CO, Gestational trophoblastic disease, In vitro fertilizatio