Endocrinol Metab > Volume 20(4); 2005 > Article
Journal of Korean Endocrine Society 2005;20(4):390-394.
DOI: https://doi.org/10.3803/jkes.2005.20.4.390    Published online August 1, 2005.
A Case of Bilateral Macronodular Adrenocortical Hyperplasia Accompanied by Hyperresponsiveness to Vasopressin.
Deok In Kim, Seung Hee Lee, Eun A Kim, Hwi La Park, Gyeong Ug Lee, Seong Bin Hong, Moon Suk Nam, Seok Hwan Shin, Yong Seong Kim
Department of Internal Medicine, Inha University, College of Medicine, Incheon, Korea, Department of Surgery, Korea.
Abstract
Cushing's syndrome associated with nodular adrenal hyperplasia glands is divided into 4 main categories: adrenal adenoma, adrenal carcinoma, primary pigmented nodular adrenal dysplasia and macronodular adrenal hyperplasia(MAH). The mechanism of bilateral MAH, when ACTH is suppressed, was previously unknown, and referred to as being "autonomous". Recently, several reports have shown MAH to be under the control of ectopic or eutopic membrane hormone. Here, a case of Cushing's syndrome, caused by bilateral MAH, is reported. A 62-year-old woman presented with Cushingoid features, hypertension and diabetes mellitus. In her case, abnormal adrenal stimulation of cortisol secretion in response to exogenous vasopression stimulation was shown. Her urine free cortisol was 726.0microgram/dL, which was not suppressed after administration of high-dose dexamethasone. Her plasma cortisol level was elevated, but without circadian rhythm. ACTH was undetectable. An abdomen CT scan demonstrated bilaterally enlarged multinodular adrenal glands. A Sella MRI revealed no alteration of the pituitary gland. The patient underwent a laparoscopic bilateral adrenalectomy. Histological examination revealed bilateral macronodular hyperplasia. After having recovered, the patient showed progressive regression of the Cushingoid status.
Key Words: Cushing's syndrome, Macronodular adrenocortical hyperplasia, Hyperresponsiveness to vasopressin, Laparoscopic bilateral adrenalectomy


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