A 39-year-old man with occult eutopic corticotropin microadenoma leading to Cushing's syndrome was presented. Magnetic resonance imaging was unable to identify the pituitary microadenoma. In another department bilateral adrenalectomy was justified. In our department to identify or exclude a pituitary source of autonomous corticotropin secretion desmopressin test was performed. Intravenous desmopressin administration enhanced both circulating concentrations ofACTH and cortisol by 217.36% (peak vs. baseline) and 67% respectively. The patient underwent a transsphenoidal adenomectomy. Immunostaining demonstrated ACTH in adenoma cells. After surgery cortisol levels returned to the normal range, moreover no ACTH and cortisol response was elicited by desmopressin administration. This clinical case represents the demonstration of the possibility to unmask an occult eutopic Cushing's syndrome with the desmopressin test.
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