Cushing's syndrome with cortisol hypersecretion from one of bilateral adrenocortical adenomas: Report of a case

Abstract

We report herein the case of a 40-year-old man with Cushing's syndrome, diagnosed by clinical manifestations and endocrinological studies, who was found to have bilateral adrenocortical adenomas, one of which hypersecreted cortisol. The Cushing's syndrome was therefore attributed to primary adrenocortical disease, and the right adrenal tumor was resected and histologically diagnosed as a so-called black adenoma. After resection of the right tumor, the left adrenal tumor showed no signs of cortisol hypersecretion for the 23 months of follow-up until the patient died of peritonitis subsequent to the rupture of a duodenal ulcer. The left adrenal tumor was examined at autopsy and found to be a cortical adenoma. These data imply that the adrenal adenomas developed primarily from the adrenal gland itself, and that one of the tumors was well differentiated and secreted excess hormones, while the other remained in cell proliferation without hypersecretion.

A Rare Case of Cushing's Syndrome due to Bilateral Adrenocortical Adenomas.

“…Immunohistochemical examination of the nodules has revealed intense activity of 3f3-hydroxysteroid dehydrogenase (3f3-HSD) in BAA, but only moderate to weak activity of 313-HSD in AIMAH [21]. In the present case, the three nodules were not entirely encapsulated, but they expressed intense activity for 3/3-HSD and the adjacent nonnodular area was atrophied with a low expression of DHEA-ST, which is a good functional by high plasma levels of cortisol [18]. Similarly, the uptake of 131I-adosterol by both adrenal glands in this patient may not confirm hypersecretion of cortisol from both adrenal glands, because a nonfunctioning adenoma can also show an increased uptake [23].…”

“…At this time, the serum PRL level was high (79.5 ng/ml), probably because she received histamine-2 receptor blocker. [5][6][7][8][9][10][11][12][13][14][15][16][17][18][19][20]. The present case is the 18th.…”

“…Adrenal venous sampling is required to determine which nodule (and which adrenal gland) actually produced the excess amount of cortisol. If there is laterality of cortisol secretion, unilateral adrenalectomy or tumorectomy (unilateral or bilateral) should be chosen to avoid the risk of hypoadrenalism produced by bilateral adrenalectomy [17,18]. Although PAC was high prior to the surgery, the possibility of aldosterone production from the adenomas was excluded because of no detectable aldosterone concentrations in the nodules [4].…”

A Patient With Preclinical Cushing's Syndrome and Excessive DHEA-S Secretion Having Unilateral Adrenal Carcinoma and Contralateral Adenoma.

“…Cases of full blown Cushing's syndrome due to bilateral adrenal tumours have been reported [13][14][15][16][17][18][19], but not in those with preclinical Cushing's syndrome as in this case. In addition, the case reported here is also the first reported case of unilateral adrenocortical carcinoma associated with contralateral adenoma.…”

Unusual causes of Cushing's syndrome

“…Bilateral adrenalectomy necessitating lifelong steroid replacement is the treatment of choice in PPNAD and AIMAH while in bilateral adenomas an effort to preserve the adrenocortical function could be made through bilateral partial adrenalectomy. It is important to document functionality of both adenomas before undergoing bilateral adrenalectomy, since there is also the possibility of the coexistence of a functional and a contralateral nonfunctioning adenoma, as has been reported in a few cases (43,44). In this case adrenal scintigraphy and/or adrenal vein sampling might be of value.…”