BACKGROUND: Epidemiological data on obstetric and oncologic complications in twin pregnancies combining a complete hydatidiform mole (CHM) coexisting with a normal fetus and placenta are limited. OBJECTIVES: To evaluate perinatal and obstetric outcomes for mother and fetus and risk of gestational trophoblastic neoplasia (GTN) in twin pregnancies including a CHM. SEARCH STRATEGY: PubMed, MEDLINE and EMBASE and the grey literature were searched for articles published between May 1980 and May 2019 using a protocol designed a priori and registered on PROSPERO (CRD42018112524). SELECTION CRITERIA: Observational cohort studies of four or more cases confirmed by histopathology and providing data on pregnancy outcomes and GTN. DATA COLLECTION AND ANALYSIS: Two reviewers independently reviewed abstracts and full-text articles. The quality of the studies was assessed with the Newcastle-Ottawa scale and a meta-analysis was performed. MAIN RESULTS: Of the 344 abstracts identified, 14 studies (244 cases) met the eligibility criteria. The incidence of maternal complication in ongoing pregnancies was 80.8% and included vaginal bleeding, hyperthyroidism and pre-eclampsia. There were overall 91 (50%) live births in ongoing pregnancies and 83 (34%) of the total cases were subsequently diagnosed with GTN. Substantial and significant (P < 0.001) heterogeneity was found for the incidence of preeclampsia indicating variability in reporting the incidence of some obstetric complications between studies. CONCLUSIONS: Patients diagnosed with a twin pregnancy combining a CHM and an apparently normal fetus have a high risk of perinatal complications, low live-birth rates and around a third of them will develop a GTN and should be managed by specialised multidisciplinary teams. TWEETABLE ABSTRACT: Our study indicates a high rate of obstetric and oncologic complications in patients presenting with a complete hydatidiform mole and coexistent normal fetus.
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