Cushing's syndrome due to unilateral nodular adrenal hyperplasia: a new pathophysiological entity?

Abstract
A case is presented of a 43-yr-old Caucasian female with Cushing''s syndrome, malignant hypertension and renal insufficiency. Plasma cortisol values were elevated (19.5 .mu.g/100 ml at 0800 h and 18.5 .mu.g/100 ml at 2200 h) with loss of diurnal variation and failure of suppression with low- and high-dose dexamethasone. 17-Ketogenic steroid excretion remained unchanged after metyrapone administration. Adrenal vein plasma cortisol values showed a prominent left-sided gradient. Gross, histologic and EM examination of the left adrenal gland revealed changes of nodular adrenal hyperplasia. The presence of a suppressed right adrenal gland was confirmed post-operatively by a 5-day ACTH infusion. This case may represent a previously unreported variant of Cushing''s syndrome due to unilateral nodular adrenal hyperplasia.

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