INTRODUCTION: Pituitary adenomas are the primary cause of Cushing's disease (CD), yet they can produce negative MRI findings when coexisting with empty sella syndrome, complicating diagnosis. This case illustrates how advanced imaging and endocrine testing overcome this diagnostic challenge. CASE DESCRIPTION: A 54-year-old woman was admitted to our hospital due to facial plethora, hyperglycemia and forearm pustules. The endocrine function test suggested CD at first. However, the initial enhanced MRI revealed an empty sella. Based on the positive results of bilateral inferior petrosal sinus sampling, we performed high-resolution dynamic enhanced MRI of pituitary gland. Finally, we discovered pituitary nodules on the background of empty sella. The patient accepted surgical treatment and the postoperative outcomes were favorable. CONCLUSION: Empty sella does not exclude the diagnosis of CD. Standardized endocrine function tests and advanced imaging technology play decisive roles in the qualitative and etiological diagnosis of CD, and we also emphasize the importance of clinical thinking.
