Cushing’s syndrome which is an uncommon but lethal disorder occurs due to many causes. The most common endogenous cause is Cushing’s disease or pituitary Cushing. The etiology of this disorder is an adenoma in the anterior pituitary. The most important point in the treatment of this disorder is differentiation between Cushing’s disease (pituitary) and Cushing’s syndrome (adrenal). We can differentiate up to 90% of cases with hormonal test and radiologic anatomy .The incidence ratio of the disorder in females to males is approximately 5:1. Bilateral adrenal hyperplasia, which is seen in Cushing’s disease, is caused by long adrenocorticotropic hormone (ACTH) secretion. This long acting secretion may cause formation of nodules in adrenals. Sometimes these unilateral nodules may cause misdiagnosis. In this article, two cases of cushing’s disease which had been misdiagnosed and led to unilateral adrenalectomy are reported. These surgeries were not effective and after proper hormonal tests approving occurrence of Cushing’s disease, both cases had Trans-Sphenoidal Surgery (TSS). After surgery the symptoms in the cases decreased. But both patients had a recurrence of the disease. Now, by means of radiotherapy and medical adrenalectomy (ketoconazole treatment) both of them are under control.