Endocrine Abstracts

Introduction: Even with a high incidence of infertility owing to the suppressive effects of hypercortisolism in the gonadal axis, either associated or not to hyperandrogenism and ovulatory dysfunction, Cushing’s syndrome (CS) during pregnancy has been reported. This could be due to a greater rate of CS among women in reproductive age; however, it is a rare condition, with fewer than 200 cases reported in the literature. In general, CS affects 2–25 people to every 1 000 000 per year. Although most cases during pregnancy are caused by autonomous cortisol secretion of adrenal glands, there are reports of Cushing’s disease (CD) within this population, providing unfavorable mother-fetus outcomes. We present a case of spontaneous pregnancy in a patient in remission of CD, that recurred during peripartum period.

Case report: A 38-year-old patient presented to the endocrinology clinic with a recent diagnosis of type 2 diabetes mellitus and systemic arterial hypertension. After the initial workup, CD was diagnosed and promptly treated with transsphenoidal surgery. In the following years there was no sign of recurrence and, five years later, the patient became pregnant. In the first trimester of pregnancy, there was a worsening of blood glucose levels and insulin has been prescribed. The patient’s condition did not improve and led to premature birth at 31 weeks of pregnancy. She remained with hyperglycemia, elevated blood pressure and asthenia, accompanied by flank purple striae, facial plethora and increased fat in the cervical area, up until two months post-partum. Laboratory workup after obstetric hospital discharge revealed increased late-night salivary cortisol of 9.71 ng/dl (0.8–1.2 ng/dl) and a 7.8% glycated hemoglobin. Plasma cortisol after 1 mg of dexamethasone was 12.2 mg/dl and ACTH 30.7 and 35 pg/ml. A new exam showed a pituitary microadenoma and the patient was again referred to the Neurosurgery service.

Conclusions: CS is a rare condition in pregnancy due to hypercortisolemia-induced infertility. Moreover, clinical features of CS can overlap physiological changes during the peripartum period; therefore, it is essential a high suspicion of recurrence in pregnant patients with a history of CS. Medical treatment is mandatory as deleterious peripartum outcomes may occur. Neurosurgery in the second trimester is the standard CD treatment in pregnancy. Cabergoline may be used, even though case reports with medical therapy alternatives are scarce. Our patient unfortunately had complications, without proper treatment during pregnancy because of a low clinical suspicion by the obstetrics team.