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A HIGH-RISK PREGNANCY IN A PATIENT WITH SWYER SYNDROME: A CASE REPORT AND REVIEW OF THE LITERATURE

Fertility and sterility(2023)

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摘要
We aim to characterize the high-risk pregnancy course, delivery, and outcome from a patient with Swyer Syndrome who underwent in-vitro fertilization (IVF) through this case report. In addition, a review of all reported pregnancies in patients with Swyer Syndrome was conducted to attempt to identify associated pregnancy outcomes. Verbal consent was obtained from the patient. We describe a 38 year-old patient with Swyer Syndrome with a history of Crohn’s disease, Stage IB dysgerminoma status post four cycles of chemotherapy, and a complicated surgical history after a Roux-en-Y gastric bypass. Her obstetric course was notable for Class II obesity, pre-viable preterm premature rupture of membranes (PPROM) at 21 weeks gestational age and a subsequent extended antepartum admission complicated by the development of gestational hypertension and COVID-19 infection. She underwent an emergency cesarean delivery at 27w5d due to placental abruption. The neonate is currently receiving ongoing neonatal intensive care. For the literature review, PubMed was queried and n=103 studies resulted. Inclusion criteria included any report of karyotype-confirmed partial or pure 46 XY gonadal dysgenesis (Swyer syndrome) who carried a pregnancy and delivered a live neonate. Exclusion criteria included reports published in languages other than English, reports excluding details regarding antenatal course or delivery, or anything out of scope of the objective. The references of the included studies were reviewed in an effort to ensure no reports were missed. Twenty-six (n=26) studies met criteria for inclusion, encompassing 44 total pregnancies (38 live deliveries, 5 miscarriages, 1 ongoing pregnancy at time of publication) over 33 years (1988-2021). Descriptive statistics were performed by the study team via Microsoft Excel. Approximately 87% of the cohort underwent cesarean delivery; 84.8% of these cesarean deliveries were attributed to routine obstetric indications. Review of the literature previously reported a 24% prevalence in hypertensive disorders of pregnancy, in this updated review the prevalence of hypertensive disorders was found to be 36.8% (n=14). This is compared to a general population prevalence of 14.6% We report on a highly complex, rare case of the pregnancy, antenatal, and delivery course of a patient with 46 XY gonadal dysgenesis (Swyer syndrome). A review of the literature demonstrates a higher than previously reported incidence of hypertensive disorders of pregnancy in these patients.
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