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We report a rare case of Cushing's syndrome induced by an ectopic adrenocortical adenoma. A 57-year-old woman was diagnosed with adrenocorticotropic hormone (ACTH)-independent Cushing's syndrome based on clinical manifestation and laboratory information. She was found to have a mass in the left renal hilum via contrast-enhanced computed tomography (CT). The mass was negative, as seen in somatostatin receptor imaging with 99mTc-hydrazinonicotinyl-Tyr3-octreotide (HYNIC-TOC), and showed mild fluorodeoxyglucose (FDG) activity via positron emission tomography (PET)/CT. The results of adrenal venous sampling suggested a left-side adrenal origin of hypercortisolism, possibly secreted by the mass in the renal hilum. Histopathology after surgical resection of the mass confirmed an ectopic adrenocortical adenoma, which was responsible for the patient's Cushing's syndrome. During the 8-month follow-up after surgery, no recurrence of Cushing's syndrome was found. [ABSTRACT FROM AUTHOR]