Abstract
A dichorionic twin pregnancy with complete hydatidiform mole and coexistent fetus is a rare and challenging situation, whose pathogenesis has not been yet fully understood. We present a case of a 39-year-old woman who underwent intracytoplasmic sperm injection with two embryos transfer. The 12-week gestation ultrasound examination revealed normal fetus and placenta with features of hydatidiform mole, leading to pregnancy termination. Autopsy and histological examinations diagnosed a complete mole coexisting with a normal fetus, and the genetic analysis showed a diploid fetus with biparental genome and molar tissue with paternal diploidy. This case highlighted that complete molar pregnancies may still occur even though pregnancy is achieved after intracytoplasmic sperm injection. A review of the literature was performed by collecting data from the few similar reported cases and by commenting on the pathogenesis of this rare condition.
Highlights
Estimates from studies conducted in North America, Australia, New Zealand, and Europe have shown that the incidence of hydatidiform mole ranges from 0.57 to 1.1 per 1000 pregnancies, whereas others in Southeast Asia and Japan have described an incidence as high as 2.0 per 1,000 pregnancies[1].Hydatidiform mole coexisting with live fetuses is a rare event with a reported incidence of 1 in 20,000 to 100,000 pregnancies[2,3]
Partial hydatidiform mole or twin pregnancy, with complete hydatidiform mole coexisting with a normal fetus (CHMCF), falls into this category
Few cases of CHMCF have been reported over the last two decades. These pregnancies are often associated with severe maternal complications, such as persistent vaginal bleeding, thromboembolic disease, severe preeclampsia, and persistent gestational trophoblastic neoplasia (GTN)[2,5,16]
Summary
Estimates from studies conducted in North America, Australia, New Zealand, and Europe have shown that the incidence of hydatidiform mole ranges from 0.57 to 1.1 per 1000 pregnancies, whereas others in Southeast Asia and Japan have described an incidence as high as 2.0 per 1,000 pregnancies[1]. Hydatidiform mole coexisting with live fetuses is a rare event with a reported incidence of 1 in 20,000 to 100,000 pregnancies[2,3]. In many instances, these pregnancies are associated with significant maternal and fetal complications including preeclampsia, thromboembolic disease, hyperemesis, hemorrhage, and intrauterine demise. Partial hydatidiform mole or twin pregnancy, with complete hydatidiform mole coexisting with a normal fetus (CHMCF), falls into this category. A few cases of CHMCF after intracytoplasmic sperm injection (ICSI) have been reported, mostly in the Asian population[6,7,8,9,10,11,12,13,14,15]. This case reported an achieved pregnancy after ICSI of a CHMCF with comprehensive genetic analysis
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