ABSTRACT
Primary pigmented adrenocortical disease is a rare disorder usually affecting both adrenals. It causes Cushing’s syndrome that is adrenocorticotropic hormone independent. It is treated by bilateral adrenalectomy. We present an unusual case where this condition was unilateral and was diagnosed as adenoma on imaging. The patient was subsequently treated by unilateral adrenalectomy, and had no signs of recurrence in 5-year postoperative follow-up. This case emphasizes the importance of histopathology and immunohistochemistry in diagnosis of this condition.