CUSHING'S SYNDROME IN PREGNANCY—TREATMENT WITH METYRAPONE

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Abstract
A 23 yr old female was presented with severe Cushing''s syndrome in the 23rd wk of pregnancy. Investigations showed plasma cortisol 770 nmol/l (0800 h) and 850 nmol/l (2300 h); plasma ACTH was 10 ng/l (0800 h) and 27 ng/l (2300 h); urinary free cortisol excretion was 2460 nmol/24 h. Dexamethasone 2 mg/6 h for 48 h suppressed the 0800 h plasma cortisol only to 680 nmol/l. Abdominal computer tomography scan showed a right adrenal adenoma. The patient was treated with metyrapone and a good clinical improvement ensued. Plasma cortisol was reduced to 300-500 nmol/l. Despite ultrasonographic evidence of normal fetal growth, urinary estriol excretion was markedly deficient. Prior to the spontaneous onset of labor, there was a marked rise in plasma cortisol despite continuous metyrapone treatment. A normal female infant was born at 37 wk gestation. The maternal adrenal adenoma was subsequently removed. The deficiency of estriol synthesis during the pregnancy may be explained by metyrapone-induced inhibition of C19-hydroxylation.

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