title: Term delivery of a complete hydatidiform mole with a coexisting living fetus followed by successful treatment of maternal metastatic gestational trophoblastic disease.
abstract: OBJECTIVE
  A twin pregnancy consisting of a complete hydatidiform mole with a coexisting normal fetus is extremely rare with an incidence of 1/22,000 to 1/100,000. The incidence of preterm delivery is high and few pregnancies reach near term with a viable fetus.
  
  
  METHODS
  A 34-year-old woman presented at 20 weeks of gestation with increased levels of serum beta human chorionic gonadotropin (beta-HCG) at 4.74 multiples of the median (310277.7 mIU/mL). Ultrasonography showed a hydatidiform mole together with a normal fetus. Fetal karyotyping revealed 46XY. The serum beta-HCG levels were followed up throughout the remainder of the pregnancy. A male infant weighting 2260 g and the molar tissue were delivered at 37 weeks of gestation. The karyotype of the molar tissue showed 46XX and the histopathological report confirmed our diagnosis. At 4 months postpartum, metastatic gestational trophoblastic disease of the lung was diagnosed in the mother by a computed tomography scan due to increased beta-HCG levels. The patient received three unsuccessful cycles of methotrexate and folinate. Another four cycles of chemotherapy consisting of etoposide, methotrexate, actinomycin D, cyclophosphamide, and vincristine (EMA-CO) were initiated and the beta-HCG levels returned to normal. There was no evidence of recurrence in the subsequent 5 years of regular follow up.
  
  
  CONCLUSIONS
  A pregnancy with a complete hydatidiform mole and a living cotwin can be a serious threat to the health of both the mother and the fetus. Early diagnosis depends on a combination of detecting an unusually high level of serum beta-HCG and ultrasound examination. We suggest that continuation of the pregnancy may be an acceptable option and that the pregnancy may continue until term if a normal fetal anatomy is assured and maternal complications are under control. Patients require careful postpartum follow up and any recurrent disease should be managed aggressively.
authors.affiliation: Department of Obstetrics and Gynecology, Chang Gung Memorial Hospital, Linkou Medical Center and Chang Gung University College of Medicine, Kwei-Shan, Taoyuan, Taiwan.
authors.forename: Hsiu-Huei
authors.initials: HH
authors.lastname: Peng
authors.affiliation: Department of Obstetrics and Gynecology, Chang Gung Memorial Hospital, Linkou Medical Center and Chang Gung University College of Medicine, Kwei-Shan, Taoyuan, Taiwan. Electronic address: kghuang@ms57.hinet.net.
authors.forename: Kuan-Gen
authors.initials: KG
authors.lastname: Huang
authors.affiliation: Department of Obstetrics and Gynecology, Chang Gung Memorial Hospital, Linkou Medical Center and Chang Gung University College of Medicine, Kwei-Shan, Taoyuan, Taiwan.
authors.forename: Ho-Yen
authors.initials: HY
authors.lastname: Chueh
authors.affiliation: Department of Obstetrics and Gynaecology, University Malaya Medical Centre, Faculty of Medicine, University of Malaya, Kuala Lumpur, Malaysia.
authors.forename: Aizura-Syafinaz
authors.initials: AS
authors.lastname: Adlan
authors.affiliation: Department of Obstetrics and Gynecology, Chang Gung Memorial Hospital, Linkou Medical Center and Chang Gung University College of Medicine, Kwei-Shan, Taoyuan, Taiwan.
authors.forename: Shuenn-Dyn
authors.initials: SD
authors.lastname: Chang
authors.affiliation: Department of Obstetrics and Gynecology, Chang Gung Memorial Hospital, Linkou Medical Center and Chang Gung University College of Medicine, Kwei-Shan, Taoyuan, Taiwan.
authors.forename: Chyi-Long
authors.initials: CL
authors.lastname: Lee
mesh_terms: D000328:Adult; D005260:Female; D006801:Humans; D006828:Hydatidiform Mole / Q000188:drug therapy / Q000473:pathology*; D007231:Infant, Newborn; D008175:Lung Neoplasms / Q000188:drug therapy / Q000556:secondary*; D008297:Male; D011247:Pregnancy; D059285:Pregnancy, Twin; D047929:Term Birth; D014594:Uterine Neoplasms / Q000188:drug therapy / Q000473:pathology*
keywords: EMA-CO; metastatic gestational trophoblastic disease; twin pregnancy with one complete hydatidiform mole