Variant of Pre-Clinical Cushing's Syndrome: Hypertension and Hypokalemia Associated with Normoreninemic Normoaldosteronism.

Yamakita, Noriyoshi; Murai, Toshihiro; Miyamoto, Koji; Matsunami, Hidetoshi; Ikeda, Tsuneko; Sasano, Hironobu; Mune, Tomoatsu

doi:10.1291/hypres.25.623

Cited by 7 publications

(6 citation statements)

References 35 publications

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“…The expression of HSD3B2, 17a-hydroxylase (CYP17A1), CYP11B1, CYP11B2, CYP11A1, CYP21A2, and DHEA-ST mRNAs was studied in tumorous and non-neoplastic tissues of four cases using molecular biological methods (21,28,33,37). Northern blot analysis revealed the expression of CYP11A1, CYP17A1, and CYP21A2 in both tumorous and non-tumorous tissue (21).…”

Section: Molecular Biological Examinationmentioning

confidence: 99%

“…The study showed that values obtained in A/CPA (1.9) were between the values obtained in APA (0.9G0.1) and CPA (2.3G0.5) and NAG (2.8). Another RT-PCR study was performed analyzing the ratio of CYP11B2 mRNA and glyceraldehydes 3-phosphate dehydrogenase in APA and CPA (37). It was distinguished between the tissue consisting mainly of clear cells and that comprised of compact cells.…”

Section: Molecular Biological Examinationmentioning

confidence: 99%

See 1 more Smart Citation

Aldosterone- and cortisol-co-secreting adrenal tumors: the lost subtype of primary aldosteronism

Späth

Korovkin

Antke³

et al. 2011

European Journal of Endocrinology

109

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Current guidelines suggest proving angiotensin-independent aldosterone secretion in patients with primary aldosteronism (PA). It is further recommended to demonstrate unilateral disease because of its consequence for therapy. A general screening for excess secretion of other hormones is not recommended. However, clinically relevant autonomous aldosterone production rarely originates in adrenal tumors, compromised of zona glomerulosa cells only. This article reviews published data on aldosterone-and cortisol-co-secreting tumors and shows that pre-operative diagnosis of such a lesion is beneficial for patients. Overt or subclinical glucocorticoid hypersecretion may interfere with diagnostic studies, e.g. adrenal venous sampling, screening of familial forms of PA on the basis of serum 18-hydroxy-cortisol (18-OH-F) determination, and provoke glucocorticoid deficiency after surgical removal of the tumor. In addition, knowledge from histological and molecular studies in patients with aldosterone-and cortisol-co-secreting tumors challenges some concepts of the development of adrenal autonomy. The presence of an aldosterone-and cortisol-co-secreting adrenocortical tumor should be considered if a patient has i) PA and an adenoma that is larger than 2.5 cm, ii) cortisol that is nonsuppressible with overnight low-dose dexamethasone, or iii) grossly elevated serum levels of hybrid steroids, such as 18-OH-F.

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Section: Molecular Biological Examinationmentioning

confidence: 99%

Section: Molecular Biological Examinationmentioning

confidence: 99%

Aldosterone- and cortisol-co-secreting adrenal tumors: the lost subtype of primary aldosteronism

Späth

Korovkin

Antke³

et al. 2011

European Journal of Endocrinology

109

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“…AUTONOMOUS production of both aldosterone and cortisol is found in adrenal adenoma, and there are approximately 20 cases that have been reported in the literature written in English [1][2][3][4][5][6][7][8][9][10][11]. The adenomas cause both primary aldosteronism and preclinical Cushing's syndrome.…”

mentioning

confidence: 99%

“…Generally there is only a tumor in the unilateral adrenal gland, which produces both aldosterone and cortisol inappropriately [1][2][3][4][6][7][8][9][10][11]. 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].…”

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confidence: 99%

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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“…Usually, cortisol and aldosterone are secreted by the same adenoma when endocrine findings indicate PA associated with SCS, and CT shows only one nodule in unilateral adrenal gland [4][5][6][7][8]. However, there is not always a single adenoma, so the result of CT can confuse our perception of the nidus of aldosterone or cortisol secretion.…”

Section: Discussionmentioning

confidence: 99%

Primary aldosteronism concurrent with subclinical Cushing’s syndrome: a case report and review of the literature

et al. 2020

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Background: The prevalence of primary aldosteronism concurrent with subclinical Cushing's syndrome was higher than previously thought. Through analyzing a rare clinical case, we summarized the diagnosis and management of primary aldosteronism with subclinical Cushing's syndrome. Case presentation: A 54-year-old Chinese man of Han nationality was diagnosed as having primary aldosteronism with subclinical Cushing's syndrome. An abdominal computed tomography scan revealed a mass in his left adrenal gland and a mass in his right adrenal gland. After finishing sequential adrenal venous sampling without adrenocorticotropic hormone, the result reminded us that the left and right nodules were responsible for hypercortisolism and aldosterone hypersecretion, respectively. Right and left adrenalectomy were performed successively. The pathological diagnosis was adrenocortical adenoma for both. Histological findings revealed that the right one had positive immunostaining for CYP11B2 and the left one had positive immunostaining for CYP11B1. The immunohistochemistry result helped us to confirm the diagnosis. Somatic KCNJ5 mutation (Leu168Arg) was found in the right tumor; there was no KCNJ5 mutation in the left adrenal tumor. Conclusions: We suggest that patients with primary aldosteronism should have a low-dose overnight dexamethasone suppression test to screen for hypercortisolism. It can help avoid misdiagnoses and contribute to proper understanding of the adrenal vein sampling result. Making sure of the nidus of aldosterone and cortisol secretion is crucial for the therapy of patients with primary aldosteronism and subclinical Cushing's syndrome.

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Variant of Pre-Clinical Cushing's Syndrome: Hypertension and Hypokalemia Associated with Normoreninemic Normoaldosteronism.

Cited by 7 publications

References 35 publications

Aldosterone- and cortisol-co-secreting adrenal tumors: the lost subtype of primary aldosteronism

Aldosterone- and cortisol-co-secreting adrenal tumors: the lost subtype of primary aldosteronism

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

Primary aldosteronism concurrent with subclinical Cushing’s syndrome: a case report and review of the literature

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