European journal of endocrinology · 2026
Sustained shrinkage of bilateral adrenal adenomas during long-term osilodrostat therapy.
СтатьяКлинический случайНазвание и аннотация: Перевод в очереди
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Messina Erika, Romanisio Martina, Gigante Antonio, Parola Cinzia, Cantore Lorenzo, Puglisi Soraya, Solitro Federica, Balbi Maurizio, Veltri Andrea, Terzolo Massimo, Reimondo Giuseppe
Аннотация
INTRODUCTION: Surgery remains the first-line therapy for all forms of Cushing's syndrome; medical therapy may be appropriate when surgery is not feasible or as adjunctive therapy in severe hypercortisolism.
CASE PRESENTATION: A 70-year-old woman was hospitalized for hemorrhagic shock due to massive abdominal bleeding. Abdominal computed tomography (CT) scan detected bilateral adrenal masses. Hormonal evaluation revealed adrenocorticotropic hormone-independent hypercortisolism. Given her frail condition, medical treatment was started while surgery was deferred. Metyrapone and ketoconazole were discontinued for inefficacy or hepatotoxicity. Osilodrostat was initiated and progressively titrated up to 6 mg/day, achieving sustained clinical and biochemical control of hypercortisolism at a maintenance dose of 1 mg/day. Adrenal masses began to shrink after 18 months of treatment, with progressive reduction through the 54-month follow-up. The patient developed adrenal insufficiency, requiring osilodrostat discontinuation and glucocorticoid replacement.
CONCLUSIONS: Osilodrostat showed marked clinical and biochemical efficacy. This case suggested that osilodrostat may contribute to adrenal mass reduction, warranting further investigation.
CASE PRESENTATION: A 70-year-old woman was hospitalized for hemorrhagic shock due to massive abdominal bleeding. Abdominal computed tomography (CT) scan detected bilateral adrenal masses. Hormonal evaluation revealed adrenocorticotropic hormone-independent hypercortisolism. Given her frail condition, medical treatment was started while surgery was deferred. Metyrapone and ketoconazole were discontinued for inefficacy or hepatotoxicity. Osilodrostat was initiated and progressively titrated up to 6 mg/day, achieving sustained clinical and biochemical control of hypercortisolism at a maintenance dose of 1 mg/day. Adrenal masses began to shrink after 18 months of treatment, with progressive reduction through the 54-month follow-up. The patient developed adrenal insufficiency, requiring osilodrostat discontinuation and glucocorticoid replacement.
CONCLUSIONS: Osilodrostat showed marked clinical and biochemical efficacy. This case suggested that osilodrostat may contribute to adrenal mass reduction, warranting further investigation.