Placental mesenchymal dysplasia associated with spontaneous ovarian hyperstimulation syndrome

Abstract

Placental mesenchymal dysplasia (PMD) is a rare disorder of unknown aetiology characterised by placentomegaly, serpiginous surface blood vessels and large cystic villi. Although a mimic of molar pregnancies and other entities, it can be distinguished from them by its association with intrauterine growth restriction or death of a relatively normal fetus and its characteristic histopathology. We report the case of a 20-year-old primigravida who presented at 16 weeks with heavy clotty vaginal bleeding and subsequent miscarriage. The placenta was grossly abnormal and resembled a partial molar pregnancy. PMD was confirmed by microscopic examination. Postmiscarriage, ultrasound revealed enlarged and multicystic ovaries similar to those seen in ovarian hyperstimulation syndrome (OHSS), which returned to normal a few weeks after miscarriage. This is the first report of PMD associated with OHSS, and we hypothesise that the most likely pathogenesis is ovarian stimulation from PMD-derived vascular endothelial growth factor.

Severe ovarian hyperstimulation syndrome in a spontaneous pregnancy with normal singleton fetus: A case report

“…For the first time, Davoudian (2015) reported a pregnant woman with placentomegaly and enlarged multicystic ovaries. He described Placental Mesenchymal Dysplasia (PMD) associated with spontaneous OHSS and hypothesized that the most likely pathogenesis is ovarian stimulation by PMD-derived vascular endothelial growth factors;[ 13 ] however, this was not the case in our patient.…”

Ovarian hyperstimulation syndrome following surgical removal of a complete hydatidiform mole: a case report

“…The occurrence of OHSS accompanied by spontaneous pregnancy is rare. As shown in Table 1, only 11 cases of OHSS after spontaneous pregnancy, with abnormal chorionic villi, have been reported until now, and of those, 9 cases were associated with a hydatidiform mole, one with an invasive mole, and one with placental mesenchymal dysplasia [2–12]. Among nine cases of OHSS that occurred after abortion of a hydatidiform mole, the first case was reported in 1966, and the ninth case was reported in 2015.…”

Severe Spontaneous Ovarian Hyperstimulation Syndrome With Cervical insufficiency: A Case Report

“…There are different mutations and polymorphisms identified in FSHR gene that can activate ovaries and cause OHSS or inactivate that can lead to sterility. In addition to the genes and receptors for FSH the genetic researches have indicated luteinizing/human chorionic gonadotrophin hormones [5][6][7]. Some recent researches remark anti-Müllerian hormone (AMH) signaling pathway in the pathogenesis of OHSS as it was mentioned that AMH may has an important regulatory effects on folliculogenesis [8][9][10].…”

“…As it is known that; leakage of intravascular fluids that cause ascites and haemoconcentration seen in OHSS, besides the genes mentioned above vascular endothelial growth factor (VEGF) can be a target while searching the underlying factor of OHSS [7]. Also, placental mesenchimal dysplasia associated with sOHSS which is the most likely pathogenesis of ovarian stimulation from placental mesenchimal dysplasia derived vascular endothelial growth factor [6].…”