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Background and Importance Ectopic Cushing’s Syndrome (ECS) is a low-incidence manifestation of high morbidity and mortality, which occurs with hypercortisolism caused by uncontrolled secretion of adrenocorticotropin (ACTH) associated with tumours, including pulmonary origin. The condition often presents with severe metabolic disturbances, making early diagnosis and management essential. To treat endogenous Cushing’s syndrome, drugs such as metyrapone, ketoconazole, and osilodrostat are available to control hypercortisolism. In this case, this treatment has been extrapolated to this patient to treat ECS of pulmonary origin.Aim and Objectives Effectiveness of osilodrostat, an 11 b hydroxylase inhibitor (enzyme responsible for the final step of cortisol biosynthesis in the adrenal gland) in a patient with Cushing’s syndrome secondary to ECS of pulmonary origin.Material and Methods Retrospective and descriptive study of a patient with ECS of pulmonary origin treated with osilodrostat. Data were obtained from the digital medical record. A literature review was conducted in PubMed and UptoDate.Results A 72-year-old patient was admitted with ACTH levels of 142 pg/ml (normal range: 7.2 - 63.3 pg/ml), basal cortisol levels of 97.7 mcg/dl (normal range 4.82 -19.5 mcg/dl), and potassium levels of 2.5 mEq/L (normal range: 3.5-5.1 meq/L). The patient was diagnosed with cortisol overproduction secondary to ectopic ACTH production of pulmonary origin. The Endocrinology Department contacted the Hospital Pharmacy Department for joint therapeutic management. Metopirone was ruled out due to the risk of worsening hypokalaemia, and a decision was made to initiate a request for ketoconazole as a foreign medication, since osilodrostat should be used if other alternatives fail. After requesting it, we were informed that ketoconazole was rejected due to the presence of a marketed domestic therapeutic alternative, osilodrostat. Treatment was therefore initiated with osilodrostat 20 mg/12 h concomitantly with hydroaltesone due to the risk of adrenal insufficiency due to complete inhibition of cortisol synthesis. Currently on treatment with osilodrostat 20 mg/12 h with normalised levels: cortisol 7.55 mcg/dl; Corticotropin: 79.6 pg/ml, potassium 4.5 meq/L (normal range: 3.5-5.1 meq/L). Given clinical stability, oncologic treatment has been initiated.Conclusion and Relevance Treatment with osilodrostat for pulmonary ECS has proven to be effective in controlling hypercotisolism. Coordination and cooperation between the Pharmacy and Endocrinology departments is essential for optimal therapeutic management.Conflict of Interest No conflict of interest