ACTH secreting macroadenomas and pituitary apoplexy are unusual in Cushing disease. In the few cases reported in the literature, they have been found in long term hypercortisolism. We communicate a 43 yr old woman with a 4 year evolution Cushing syndrome, who developed sudden cephalea and oftalmoplejia. A Computed Tomography of the pituitary fossa disclosed a macroadenoma with intracapsular hemorrhage and suprasellar expansion. In the functional tests, serum cortisol was suppressed with dexamethasone in a dose of 1 and 8 mg and responded to the desmopressin stimulus. Nevertheless, cortisol levels were lower than those observed in Cushing syndrome of similar magnitude. The tumor was resected by transphenoidal surgery and immunohistochemistry to ACTH was positive. In this case, the laboratory results suggest a partial remission of the hypercortisolism after pituitary apoplexy.