Endocrine Abstracts

Osilodrostat as an inhibitor of adrenal 11B-hydroxylas, is an effective medication used in the management of endogenous hypercortisolism. There are limited data regarding usage of osilodrostat in ectopic Cushing Syndrome (CS). Surgery is usually the first line treatment in ectopic CS, however the source of ectopic hormonal production often remains undiscovered for a long period of time. Meanwhile dangerous state of severe hypercortisolism needs to be managed pharmacologically by steroidogenesis inhibitors. We present a case of a 72 year- old male patient with diabetes mellitus, hypertension, obesity, dyslipidemia who was admitted to Emergency Department due to Transient Ischemic attack (TIA), severe hypokalemia and hyperglycemia. Physically patient presented Cushing Syndrome’s features. Additional biochemical assesment showed ACTH dependent hypercortisolemia (ACTH 197 pg/ml, Cortisol 76 ug/dl). Patient was transferred to Endocrinology Department. Hormonal tests suggested ectopic Cushing Syndrome. Either 2-[18F]FDG PET/CT or [68Ga]Ga-DOTA-TATE PET/CT didn’t show potential ectopic lesion. Pituitary MRI showed Rathe cyst. We put the patient on metyrapone (up to 2250 mg) and observed dropping of cortisol level. After 3 months of treatment metyrapone was withdrawn and osilodrostat was started 1mg BID, followed by dose reduction to 1mg per day. The Patient reached satisfactory clinical and biochemical control of Cushing Syndrome. Since cortisol levels dropped, we plan repetition of [68Ga]Ga-DOTA-TATE PET/CT. To sum up, osilodrostat seems to be an effective, safe and convenient treatment in management of severe hypercortisolism in patients with ectopic CS.