A Case of Aldosterone-Producing Adrenocortical Adenoma Associated with a Probable Post-Operative Adrenal Crisis: Histopathological Analyses of the Adrenal Gland

Sugawara, Akira; Takeuchi, Kazuhisa; Suzuki, Takashi; Itoi, Keiichi; Sasano, Hironobu; Ito, Sadayoshi

doi:10.1291/hypres.26.663

Cited by 22 publications

(27 citation statements)

References 22 publications

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“…Collectively, concomitant production of cortisol should be considered in PA patients with relatively larger adrenal tumors and/ or low plasma ACTH levels. In agreement with a previous report [28], hydrocortisone replacement after adrenalectomy was needed in our PA/SCS patients. Thus, one should rule out the possible association of SCS among PA patients in order to prevent postoperative adrenal insufficiency.…”

Section: Discussionsupporting

confidence: 93%

Clinicopathological features of primary aldosteronism associated with subclinical Cushing's syndrome

et al. 2011

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Primary aldosteronism (PA) is characterized by autonomous aldosterone hypersecretion from adrenal gland, resulting in low-renin hypertension often accompanied by hypokalemia and metabolic alkarosis. Recent epidemiologic studies have revealed that PA is more common cause of secondary hypertension than previously thought with its higher prevalence (5-15 %) in patients with hypertension [1][2][3][4][5]. Subclinical Cushing's syndrome (SCS) is characterized as subtle cortisol hypersecretion from adrenal tumor but lack of specific signs or symptoms of overt abstract. Primary aldosteronism (PA), an autonomous aldosterone hypersecretion from adrenal adenoma and/or hyperplasia, and subclinical Cushing syndrome (SCS), a mild but autonomous cortisol hypersecretion from adrenal adenoma without signs or symptoms of Cuhing's syndrome, are now well-recognized clinical entities of adrenal incidentaloma. However, the clinicopathological features of PA associated with SCS (PA/SCS) remain unknown. The present study was undertaken to study the prevalence of PA/SCS among PA patients diagnosed at our institute, and characterize their clinicopathlogical features. The prevalence of PA/SCS was 8 of 38 PA patients (21%) studied. These 8 PA/SCS patients were significantly older and had larger tumor, higher serum potassium levels, lower basal plasma levels of aldosterone, ACTH and DHEA-S as well as lower response of aldosterone after ACTH stimulation than those in 12 patients with aldosterone-producing adenoma without hypercortisolism. All 8 PA/SCS patients showed unilateral uptake by adrenal scintigraphy at the ipsilateral side, whereas the laterality of aldosterone hypersecretion as determined by adrenal venous sampling varied from ipsilateral (3), contralateral (2), and bilateral side (2). 6 PA/SCS patinets who underwent adrenalectomy required hydrocortisone replacement postoperatively. Histopathological analysis of the resected adrenal tumors from 5 PA/SCS patients revealed a single adenoma in 3, and double adenomas in 2, with varying degrees of positive immunoreactivities for steroidgenic enzymes (3β-HSD, P450 C17 ) by immunohistochemical study as well as CYP11B2 mRNA expression as measured by real-time RT-PCR. In conclusion, PA/SCS consists of a variety of adrenal pathologies so that therapeutic approach differs depending on the disease subtype.

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Section: Discussionsupporting

confidence: 93%

Clinicopathological features of primary aldosteronism associated with subclinical Cushing's syndrome

et al. 2011

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“…Therefore, we suggest that a dexamethasone-suppression test be performed on each patient with PA before surgery or selective adrenal venous sampling. This may be of particular importance for patients with PA because of the comparatively large adrenal adenomas because A/CPAs tend to be larger than pure APAs, 19,20,26 a tendency that held true for our patients.…”

Section: Discussionmentioning

confidence: 58%

“…The true prevalence of A/CPAs may also be underestimated because hypercortisolism is not routinely excluded in patients with PA at most institutions. However, the removal of A/CPAs is reported to result in adrenal crisis, 19 and our two patients depended on hydrocortisone replacement therapy after the operation. In addition, cortisol co-secretion by APA may also lead to the misinterpretation of hormonal ratios obtained during adrenal venous sampling because correcting the aldosterone values for autonomously secreted cortisol levels may give false-negative aldosterone-to-cortisol ratios at the side of the adenoma and a falsely low cortisol level in the contralateral vein.…”

Section: Discussionmentioning

confidence: 79%

Sporadic solitary aldosterone- and cortisol-co-secreting adenomas: endocrine, histological and genetic findings in a subtype of primary aldosteronism

et al. 2010

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Adrenal adenomas producing both aldosterone and cortisol (A/CPAs) have been described in only a few cases. Correct subtype classification is necessary for making therapeutic decisions in primary aldosteronism (PA). Therefore, we studied in detail the clinical, hormonal and histological features of this entity in two patients with A/CPAs. We describe two patients with A/CPA and present their endocrine evaluations at baseline, after suppression with fludrocortisone and dexamethasone, after therapy with spironolactone and after unilateral adrenalectomy. Moreover, the expression of corticotropin (MC2R) and angiotensin II type 1 (AT1R) receptors and 17a-hydroxylase in the tumors of these two patients was analyzed by immunohistochemistry. Aldosterone, 18-hydroxycorticosterone (18-OH-B) and 18-hydroxycortisol (18-OH-F) were not suppressible with fludrocortisone in either patient and were partly suppressible with dexamethasone in one of the patients. Adrenal insufficiency developed in both patients after operation and lasted for more than 6 months. Aldosterone and hybrid corticosteroids returned to normal 8 weeks after adrenalectomy. In both cases, immunostaining showed weak expression of AT1R and MC2R but strong expression of 17a-hydroxylase. The most common germline mutations in the aldosterone synthase gene and the aldosterone synthase/11b-hydroxylase hybrid gene were absent. These two cases document the fact that sporadic A/CPA is a subtype of PA. The presence of an A/CPA should be considered if a patient has both PA and hypercortisolism.

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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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A Case of Aldosterone-Producing Adrenocortical Adenoma Associated with a Probable Post-Operative Adrenal Crisis: Histopathological Analyses of the Adrenal Gland

Cited by 22 publications

References 22 publications

Clinicopathological features of primary aldosteronism associated with subclinical Cushing's syndrome

Clinicopathological features of primary aldosteronism associated with subclinical Cushing's syndrome

Sporadic solitary aldosterone- and cortisol-co-secreting adenomas: endocrine, histological and genetic findings in a subtype of primary aldosteronism

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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