Cushing's syndrome in pregnancy with a severe maternal complication: A case report
Department of Obstetrics and Gynecology, School of Medicine, Gyeongsang National University, Jinju, Korea.Journal of Obstetrics and Gynaecology Research (Impact Factor: 0.93). 02/2011; 37(2):163-7. DOI: 10.1111/j.1447-0756.2010.01339.x
Cushing's syndrome (CS) in pregnancy may be confused with a complication of pregnancy, such as pre-eclampsia or gestational diabetes. We managed a case of CS in pregnancy that was considered to be severe pre-eclampsia due to uncontrolled hypertension. The fetus was delivered via emergency cesarean section at 31 weeks' gestation because of severe pre-eclampsia and pulmonary edema. The parturient was admitted to the intensive care unit for severe maternal complications, including pulmonary hemorrhage, acute renal failure, disseminated intravascular coagulopathy, and congestive heart failure. A spine magnetic resonance image and 99m-technetium whole-body scan obtained postpartum showed multiple thoracolumbar spine compression fractures (Deleted; t-2,5,8,10,11, and -12; and L-1,2,3,4, and -5), multiple rib fractures, and a left iliac bone fracture due to osteoporosis. As a result of diagnosing CS after delivery, an adrenal cortical adenoma of the right adrenal gland was demonstrated and a laparoscopic adrenalectomy was successfully performed.
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ABSTRACT: Introduction During normal pregnancy there are significant changes in hypothalamic-pituitary-adrenal axis, with increased levels of plasma cortisol and adrenocorticotropic hormone which sometimes reach values observed in patients with Cushing’s syndrome. Cushing’s syndrome (CS) is rarely encountered during pregnancy, but is associated with serious maternal and fetal complications. Case presentation A 31-year-old female was admitted to our institution four weeks after delivery. Physical examination revealed moon face, purple striae throughout the abdomen, bruising over the legs, a dorsocervical fat pad and hirsutism. She delivered a eutrophic preterm newborn at 34 weeks gestation, without any maternal or fetal complications during delivery. Imaging showed a mass in the right suprarenal gland with a normal pituitary. After four weeks the patient underwent a right adrenalectomy. The mass was eventually identified as an adrenocortical adenoma. Conclusion In our case the diagnosis of CS was established only after pregnancy, which enabled the development of numerous adverse consequences secondary to increased plasma cortisol. If CS is recognized during pregnancy, treatment and its timing could be carefully chosen according to the patient’s individual characteristics.
Article: Adrenal disease in pregnancy[Show abstract] [Hide abstract]
ABSTRACT: Adrenal disorders in pregnancy are relatively rare, yet can lead to significant maternal and fetal morbidity. Making a diagnosis is challenging as pregnancy may alter the manifestation of disease, many signs and symptoms associated with pregnancy are also seen in adrenal disease, and the fetal-placental unit alters the maternal endocrine metabolism and hormonal feedback mechanisms. The most common cause of Cushing's syndrome in pregnancy is an adrenal adenoma, followed by pituitary etiology, adrenal carcinoma, and other exceedingly rare causes. Medical therapy of Cushing's syndrome includes metyrapone and ketoconazole, but generally surgical treatment is more effective. Exogenous corticosteroid administration is the most common cause of adrenal insufficiency, followed by the endogenous causes of ACTH or CRH secretion. Primary adrenal insufficiency is least common. A low early morning cortisol <3 mcg/dL (83 mmol/L) in the non-stressed state and in the setting of typical clinical symptoms confirms the diagnosis. In the second and third trimester cortisol rises to levels 2-3 fold above those in the non-pregnant state, therefore a baseline level of <30 mcg/dL (823 mmol/L) warrants further evaluation. ACTH stimulated normal cortisol values have been established for each trimester. Hydrocortisone, which does not cross the placenta, is the glucocorticoid treatment of choice, and fludrocortisone is used as mineralocorticoid replacement in patients with primary disease. Congenital adrenal hyperplasia is an autosomal recessive disorder; 21-hydroxylase deficiency (21OHD) is the most common form of the disease. Non-classical 21OHD is most common, followed by the salt-wasting and simple virilizing forms. The treatment of choice for pregnant women affected with CAH is hydrocortisone, and fludrocortisones is added for those with the salt-wasting form of the disease. If the fetus is at risk for classical CAH, dexamethasone treatment can be used prenatally to prevent masculinization of the genitalia in a female infant. Because dexamethasone crosses the placenta, it should not be used to treat pregnant women with CAH if the fetus is not at risk for the disease.
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ABSTRACT: A 32-year-old female, gravid two, para one, with Cushing’s syndrome (CS) was admitted to our hospital at 25 week of gestation with severe hypercortisolism. Basal urinary free cortisol (UFC) was elevated about 10 times above the upper limit of normal in two separate times and plasma cortisol failed to suppress after an overnight 1mg dexamethasone suppression test but Adrenocorticotropic hormone (ACTH) level was suppressed. An abdominal non-contrast magnetic resonance imaging (MRI) disclosed a 3-cm right adrenal mass (Fig. 1). Due to her critical general condition, the adrenalectomy was not performed. At 30 week of gestation, by the diagnosis of severe preeclampsia she underwent an emergent cesarean section. Two weeks later, right adrenalectomy was performed via laparotomy. Pathologic examination of the gland showed a benign adrenocortical adenoma. The newborn was a healthy male who weighed 1850 gram. There was no clinical or biochemical suppression of adrenocortical function in child and they were discharged after 40 days.