A Case Report of Cushing's Syndrome Due to Adrenocortical Nodular Dysplasia, With Remission Period for About 6 Years After Unilateral Adrenalectomy

A 29 y.o. female had been associated with Cushing's syndrome, gradually, from 1977. She was advised to get further examination at our hospital on September 1980. A pre-operative diagnosis of Cushing's syndrome due to bilateral adrenocortical tumors was made and right adrenalectomy was performed on December 22, 1980, as the 1st of two stage operation. The right adrenal, 1.8 cm in diameter, had a pigmented nodule and multiple pigmented micronodules, which were diagnosed pathologically as primary adrenocortical nodular dysplasia. It was not necessary for this patient to maintain steroid therapy, but after 3 months she complained of the withdrawal syndrome and was administered dexamethasone. Since July, 1981, she had no symptom without adrenocortical steroid administration. In November 1986, she became cushingoid and on June 1, 1987, we performed left adrenalectomy on her. The appearance of the left adrenal was similar to the right. We have found no reports of the cases of adrenocortical nodular dysplasia such as delayed appearance of withdrawal syndrome after unilateral adrenalectomy. In this case, plasma ACTH had relatively poor correlation with cortisol. Her plasma ACTH was not always suppressed and sometimes within normal range. Her adrenals had not only autonomy but also pituitary dependency. It is concluded that in this case there may have been a pituitary-adrenal dual control.