title: ACTH-independent Cushing's syndrome in pregnancy with spontaneous resolution after delivery: control of the hypercortisolism with metyrapone.
abstract: A 25-year-old primigravid woman presented with Cushing's syndrome at 23 weeks gestation; serum cortisol was 1090 nmol/l at 0900 h, 1230 nmol/l at 2200 h; basal urinary free cortisol excretion was 3680 nmol/24 h, and 8830 nmol/24 h after dexamethasone 8 mg daily for 48 hours; plasma ACTH was < 1.1 pmol/l. CT scan of the adrenal glands showed bilateral adrenal hyperplasia. The hypercortisolism was controlled with metyrapone until elective delivery of the fetus by Caesarean section at 34 weeks gestation because of a decline in growth. No adverse fetal effects of metyrapone treatment were apparent, maternal outcome was uncomplicated and wound healing was unimpaired. Maternal adrenocortical function had returned to normal within 4 weeks of the cessation of pregnancy and biochemical remission has been maintained up to 9 months post-partum. Metyrapone therapy is effective in controlling the hypercortisolism in certain cases of Cushing's syndrome complicating pregnancy.
authors.affiliation: Department of Diabetes and Endocrinology, Dudley Road Hospital, Birmingham, UK.
authors.forename: C F
authors.initials: CF
authors.lastname: Close
authors.forename: M C
authors.initials: MC
authors.lastname: Mann
authors.forename: J F
authors.initials: JF
authors.lastname: Watts
authors.forename: K G
authors.initials: KG
authors.lastname: Taylor
mesh_terms: D000324:Adrenocorticotropic Hormone / Q000097:blood; D000328:Adult; D003480:Cushing Syndrome / Q000097:blood / Q000188:drug therapy* / Q000652:urine; D036861:Delivery, Obstetric; D005260:Female; D006801:Humans; D006854:Hydrocortisone / Q000097:blood* / Q000652:urine; D008797:Metyrapone / Q000627:therapeutic use*; D011247:Pregnancy; D011248:Pregnancy Complications / Q000097:blood / Q000188:drug therapy* / Q000652:urine; D011262:Pregnancy Trimester, Second; D012075:Remission, Spontaneous
chemical_list: D000324:Adrenocorticotropic Hormone; D006854:Hydrocortisone; D008797:Metyrapone