Primary aldosteronism with cortisol overproduction from bilateral multiple adrenal adenomas.

Nagae, Akihiro; Murakami, Eiki; Hiwada, Kunio; Kubota, Osamu; Takada, Yasuharu; Ohmori, Takaaki

doi:10.2169/internalmedicine1962.30.26

Cited by 23 publications

(21 citation statements)

References 10 publications

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“…Only 10 cases have been reported in english and Japanese, as far as we can determine from the databases of PubMed, and Japana Centro Revuo Medicina. (Table III) [7][8][9][10][11][12][13][14][15]. Multiple adenomas are known to be seen occasionally in the adrenal gland with PA.…”

Section: Discussionmentioning

confidence: 99%

Cushing's Syndrome by Left Adrenocortical Adenoma Synchronously Associated with Primary Aldosteronism by Right Adrenocortical Adenoma: Report of a Case

et al. 2009

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mal range; 30-159) pg/ml and 10 (4.4-17.4) µg/dl, respectively. Although her plasma renin activity (PRA) and ACTH was suppressed to an undetectable level, no further examination was conducted or followedup. One year later, hypertension and progressive edema in the lower extremities gradually appeared. Reexamination revealed hypopotassemia and elevation of plasma aldosterone level as high as 216 (normal range; 30-159) pg/ml, with low PRA as 0.3 (0.5-2.0) ng/ml/ hr. Magnetic resonance imaging demonstrated that the right adrenal mass did not enlarge. Abstract. Synchronous associations of Cushing's syndrome (CS) and primary aldosteronism (PA) with multiple adrenocortical adenomas secreting each hormone independently have rarely been reported. Herein, we describe a unique case of PA associated with CS with detailed clinical and pathological investigations. bilateral adrenal masses with clinical symptoms of CS and PA were found in a 43-year-old woman. Venous sampling demonstrated excess secretion of cortisol, and aldosterone from right, and left tumor, respectively. A bilateral laparoscopic partial adrenalectomy was undergone. The right adrenal tumor (3 cm) was yellow in color with abundant lipofuscin granules, and was composed of both eosinophilic compact cells and clear cells. In situ hybridization showed that both mRNAs for HSd3b2 and CYP17A1 were strongly expressed in the tumor, suggesting cortisol synthesis. Left adrenal tumor (2.4 cm) was golden-yellow in color, and composed of clear cells only. expression of HSd3b2 and CYP11b mRNAs were observed in the tumor compatible with the aldosterone synthesis. Furthermore, minute nodules were found at the surface of normal-appearing cortex on both sides of the adrenal glands, and the expression of HSd3b2 and CYP11b mRNAs was clearly demonstrated within the nodules, indicating aldosterone synthesis. we diagnosed that the present case had 1) cortisol-producing right adrenocortical adenoma, 2) aldosterone producing left adrenocortical adenoma, and 3) cortical minute nodules with aldosterone production in both adrenal glands compatible with idiopathic adrenal hyperplasia. we reviewed the cases reported, and discussed the significance of the minute nodules in the adrenal cortex, often found in association with the adrenocortical adenoma.Key words: Adrenal gland, Cushing's syndrome, primary aldosteronism, bilateral adrenocortical adenomas, Laparoscopic adrenalectomy (Endocrine Journal 56: [495][496][497][498][499][500][501][502] 2009) A JApAnese woman was admitted to a local hospital for the treatment of her depression when she was age 35. Right adrenal mass of less than 20 mm was demonstrated by imaging studies during her workup. At this time, both plasma aldosterone and plasma cortisol levels were within normal limits, at 72 (nor-

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Section: Discussionmentioning

confidence: 99%

Cushing's Syndrome by Left Adrenocortical Adenoma Synchronously Associated with Primary Aldosteronism by Right Adrenocortical Adenoma: Report of a Case

et al. 2009

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“…A few of the variant cases have more than two tumors in uni-or bilateral adrenal glands, which autonomously produce both two hormones [5,15,16]. There are only two cases with concurrent primary aldosteronism and Cushing's syndrome that had double adenomas in the unilateral adrenal gland [5,17].…”

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Possibly Simultaneous Primary Aldosteronism and Preclinical Cushing's Syndrome in a Patient with Double Adenomas of Right Adrenal Gland

et al. 2007

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Abstract. We reported a rare case of simultaneous primary aldosteronism and preclinical Cushing's syndrome due to unilateral double adrenocortical adenomas in a 57 year-old woman who had had hypertension for the last 10 years. Abdominal computed tomography showed double tumors in her right adrenal gland. Physical findings revealed simple obesity and hypertension, but no other abnormal findings were detected. Laboratory findings demonstrated that serum potassium was 3.8 mmol/l; plasma renin activity, 0.3 ng/ml/h; plasma aldosterone, 100 pg/ml, and aldosterone renin ratio (ARR), 33. Serum cortisol was 15.7 µg/dl. There was no circadian rhythm of serum cortisol, and no suppression of serum cortisol in response to exogenous dexamethasone administration. Right adrenalectomy was performed under laparoscopy. Two well-circumscribed tumors, whose sizes were 21 and 19 mm in greatest diameter, were detected. They were macroscopically composed of a golden-yellow portion admixed with a brown portion, which corresponded to clear cells and compact cells, respectively. Immunohistochemical staining for steroidogenic enzymes demonstrated the presence of all the enzymes involved in corticosteroidogenesis in these two adenomas, indicating that the two adenomas produced both cortisol and mineralocorticoid. Specifically, one adenoma mainly caused excessive production of cortisol as compared to the other one. These findings indicate that overproduction of both cortisol and mineralocorticoid was evident in the two adenomas of the right adrenal gland in immunohistochemical study for steroidogenic enzymes, whereas there was less clinical manifestation of primary aldosteronism and Cushing's syndrome in the present patient.

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“…Most of the reported cases of the comorbidity can generally be classified into three groups. They comprise (i) both aldosterone and cortisol being simultaneously produced from a single, sometimes multiple, adrenocortical adenoma (A/CPA), (14)(15)(16)(17)(18)(19)(20) (ii) aldosterone produced from APA and cortisol from CPA, and these adenomas may exist unilaterally or separately in each other side, (21-27) (iii) aldosterone from both adrenal glands with bilateral hyperplasia (IHA) and cortisol from CPA (28,29).…”

Section: Discussionmentioning

confidence: 99%

A Case of Cortisol Producing Adrenal Adenoma Associated with a Latent Aldosteronoma: Usefulness of the ACTH Loading Test for the Detection of Covert Aldosteronism in Overt Cushing Syndrome

et al. 2012

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A 36-year-old woman with Cushing syndrome was evaluated for coexisting hyperaldosteronism, which was suggested by an abnormal response of the aldosterone-to-cortisol ratio in peripheral blood to the ACTHadministration despite a low basal aldosterone-to-renin ratio. Computed tomography revealed two independent tumors in the left adrenal gland, and adrenal venous sampling indicated hyperaldosteronism in addition to hypercortisolism in the same side. Postsurgical study including immunohistochemical analysis of steroidogenic enzymes suggested one adenoma to be cortisol-producing and the other, aldosterone-producing. The comorbidity of these different hormone-producing adenomas is not rare and careful pre-surgical evaluation is necessary to avoid post-surgical exacerbation of latent hyperaldosteronism.

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Primary aldosteronism with cortisol overproduction from bilateral multiple adrenal adenomas.

Cited by 23 publications

References 10 publications

Cushing's Syndrome by Left Adrenocortical Adenoma Synchronously Associated with Primary Aldosteronism by Right Adrenocortical Adenoma: Report of a Case

Cushing's Syndrome by Left Adrenocortical Adenoma Synchronously Associated with Primary Aldosteronism by Right Adrenocortical Adenoma: Report of a Case

Possibly Simultaneous Primary Aldosteronism and Preclinical Cushing's Syndrome in a Patient with Double Adenomas of Right Adrenal Gland

A Case of Cortisol Producing Adrenal Adenoma Associated with a Latent Aldosteronoma: Usefulness of the ACTH Loading Test for the Detection of Covert Aldosteronism in Overt Cushing Syndrome

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