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

Makino, Shinya; Chikazawa, Hiroaki; Yorimitsu, A; Suehiro, Tadashi; Hashimoto, Kozo; Ohashi, Yozo; Morioka, Motohiro; Sasano, H

doi:10.1507/endocrj.44.533

Cited by 9 publications

(4 citation statements)

References 12 publications

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“…Although the diagnosis and management of corticotropin (ACTH)-independent Cushing syndrome (CS) or subclinical CS in patients with a unilateral cortical adenoma is relatively straightforward [1,2], the management of patients with bilateral adrenal masses in these settings is problematic [3][4][5][6][7][8][9][10][11][12]. These patients may have a unilateral cortisol-secreting adenoma with a contralateral nonfunctioning cortical adenoma, bilateral cortisol-secreting adenomas, or bilateral ACTH-independent macronodular adrenal hyperplasia (AIMAH) masquerading as bilateral single adenomas.…”

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confidence: 99%

The Clinical Conundrum of Corticotropin‐independent Autonomous Cortisol Secretion in Patients with Bilateral Adrenal Masses

et al. 2007

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The Clinical Conundrum of Corticotropin‐independent Autonomous Cortisol Secretion in Patients with Bilateral Adrenal Masses

et al. 2007

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“…Bilateral cortisol-secreting adenomas are a rare cause of CS, a condition that is predominantly seen in women of child-bearing age [2][3][4][5][6] as was the case in our patient. Making a differential diagnosis between cortisol-secreting adenomas and ACTH-independent macronodular adrenal hyperplasia (AIMAH) or unilateral cortisol-secreting adenoma with a contralateral nonfunctioning cortical adenoma is a challenge [12].…”

Section: Discussionmentioning

confidence: 62%

“…Bilateral cortisol-secreting adenomas, however, are extremely rare [2][3][4][5][6]. In contrast to ACTHdependent CS and adrenocortical-carcinomas, adrenocortical adenomas generally (with rare exception [7]) only secrete glucocorticoids without co-secretion of adrenal androgens [8].…”

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A case of Cushing’s syndrome due to bilateral cortisol-secreting adenomas with unilateral DHEAS oversecretion

et al. 2015

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Cortisol-seCreting adrenal adenomas account for 10-15% of endogenous Cushing's syndrome (CS) cases [1]. Bilateral cortisol-secreting adenomas, however, are extremely rare [2][3][4][5][6]. In contrast to ACTHdependent CS and adrenocortical-carcinomas, adrenocortical adenomas generally (with rare exception [7]) only secrete glucocorticoids without co-secretion of adrenal androgens [8]. We herein report a rare case of CS due to bilateral cortisol-secreting adrenal adenomas (one on the left and two on the right) with unilateral (left-sided) dehydroepiandrosterone sulphate (DHEAS) oversecretion. This is a unique hormonal Abstract. A 39-year-old Japanese woman presented with typical clinical symptoms of Cushing's syndrome, including amenorrhea and hirsutism, for 2 years. The results of her initial endocrine evaluation were consistent with ACTHindependent Cushing's syndrome due to bilateral adrenal masses (diameter of 3.1 cm and 2.4 cm on the left and right, respectively). Serum dehydroepiandrosterone levels were 6,901 ng/mL (normal range 230-2,660 ng/mL). Bilateral laparoscopic adrenalectomies were performed (left adrenalectomy first and right adrenalectomy 3 months later). Following the left adrenalectomy, the results of the endocrine evaluations were still consistent with a diagnosis of ACTH-independent Cushing's syndrome. Serum dehydroepiandrosterone sulphate levels, however, were below the normal range (143 ng/mL). Unexpectedly, the patient's menstruation resumed 2.5 months after the left adrenalectomy. Pathological examination of the resected glands showed bilateral adrenocortical adenomas, one on the left with a diameter of 3 cm, and two on the right with diameters of 0.7 cm and 1.3 cm. Immunohistochemical analysis revealed side chain cleavage, 17α-hydroxylase, 3β-hydroxysteroid dehydrogenase, and 21-hydroxylase immunoreactivity in each adenoma. Dehydroepiandrosteronesulfotransferase immunoreactivity was pronounced in the left adenoma, less pronounced in one of the right adenoma and weak in the other right adenoma. These results were consistent with clinical endocrine findings. Ours is the first case of a patient with bilateral cortisol-secreting adenomas with unilateral oversecretion of dehydroepiandrosterone sulphate. Resumption of menstruation after the correction of the dehydroepiandrosterone-sulphate excess, despite persistent cortisol excess, indicates the importance of adrenal androgens for the regulation of the menstrual cycle.

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“…In BAA, the nodules are usually encapsulated and the adjacent non-nodular area is atrophic, whereas in AIMAH the nodules are not encapsulated, and the adjacent area is hypertrophic and sometimes contains small micronodules. 10 There are four approaches to laparoscopic adrenalectomy, namely, anterior transperitoneal, lateral transperitoneal, lateral retroperitoneal, and posterior retroperitoneal. We used the lateral transperitoneal approach in our patient because it offered good visibility of familiar anatomic landmarks, easy access to other organ systems, the use of gravity to retract the spleen and liver, and a wide exposure to allow easy removal of large adrenal lesions.…”

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confidence: 99%

Laparoscopic Bilateral Partial Adrenalectomy for Adrenocortical Adenomas Causing Cushing's Syndrome: Report of a Case

et al. 2005

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Laparoscopic total adrenalectomy has become a standard technique for small adrenal tumors; however, bilateral adrenalectomy results in postoperative adrenal insufficiency, necessitating lifelong steroid replacement. To preserve adrenocortical function in a 41-year-old woman with bilateral adrenocortical adenoma (BAA) causing Cushing's syndrome, we performed laparoscopic bilateral partial adrenalectomy. We based our preoperative diagnosis of bilateral adrenocortical tumors causing Cushing's syndrome on the results of endocrinological investigations and imaging findings. Thus, we performed lateral transperitoneal laparoscopic bilateral partial adrenalectomy, preserving the adrenal glands, which were normal. Pathological examination of both tumors confirmed the diagnosis of adrenocortical adenoma. The patient had no postoperative complications, and her adrenocortical function was normal without steroid replacement at her 10-month follow-up. This report shows that Cushing's syndrome resulting from bilateral adenomas can be effectively treated by laparoscopic bilateral partial adrenalectomy as a minimally invasive, adrenocortical-preserving operation.

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

Cited by 9 publications

References 12 publications

The Clinical Conundrum of Corticotropin‐independent Autonomous Cortisol Secretion in Patients with Bilateral Adrenal Masses

The Clinical Conundrum of Corticotropin‐independent Autonomous Cortisol Secretion in Patients with Bilateral Adrenal Masses

A case of Cushing’s syndrome due to bilateral cortisol-secreting adenomas with unilateral DHEAS oversecretion

Laparoscopic Bilateral Partial Adrenalectomy for Adrenocortical Adenomas Causing Cushing's Syndrome: Report of a Case

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

Cited by 9 publications

References 12 publications

The Clinical Conundrum of Corticotropin‐independent Autonomous Cortisol Secretion in Patients with Bilateral Adrenal Masses

The Clinical Conundrum of Corticotropin‐independent Autonomous Cortisol Secretion in Patients with Bilateral Adrenal Masses

A case of Cushing&rsquo;s syndrome due to bilateral cortisol-secreting adenomas with unilateral DHEAS oversecretion

Laparoscopic Bilateral Partial Adrenalectomy for Adrenocortical Adenomas Causing Cushing's Syndrome: Report of a Case

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A case of Cushing’s syndrome due to bilateral cortisol-secreting adenomas with unilateral DHEAS oversecretion