A case of long-standing Cushing's syndrome with an unusual form of bilateral multinodular adrenal hyperplasia has been studied. The patient demonstrated hyperresponsiveness to ACTH, and failure of suppression to a prolonged course of dexamethasone. Steroid excretion was characterized by high levels of 3β-hydroxy-Δ6-steroids and cortisol metabolites, but not tetrahydro S (3α,17,21-trihydroxy-5β-pregnan-20-one). Possible relationships to adenoma or carcinoma are considered.

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