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Bilateral macronodular adrenal hyperplasia (BMAH) is a rare cause of adrenal Cushing's syndrome, accounting for less than 1% of cases. We here report the case of a 48-year-old patient with diabetes and hypertension presenting with clinical signs of Cushing's syndrome. Etiological assessment enabled clinicians to retain the diagnosis of Adrenocorticotropic hormone (ACTH)-independent hypercortisolism associated with BMAH. Unilateral left adrenalectomy was performed based on noriodocholesterol scintigraphy results, with good outcome. However, given the risk of recurrence and cardiovascular complications, long-term monitoring was scheduled. [ABSTRACT FROM AUTHOR]