We report the case of a young woman presenting with a plurihormonal pituitary macroadenoma first secreting prolactin and leading to a galactorrhea/oligomenorrhea syndrome, which was treated with dopamine agonists, and later inducing an overt ACTH-dependent Cushing syndrome diagnosed during early pregnancy. This evidence concerns the gene POMC and Cushing syndrome due to macronodular adrenal hyperplasia.