Overt Diabetes Mellitus in a Patient with Combined Primary Aldosteronism and Cushing's Syndrome

Sasaki, Nobuhiro; Iwase, Masanori; Arima, Hisatomi; Nohara, Satoshi; Bandai, Sachiko; Yao, Takashi; Fujii, Koji; Iida, Mitsuo

doi:10.2169/internalmedicine.45.1855

Cited by 8 publications

(4 citation statements)

References 17 publications

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“…Many cases of PA with hypercortisolism reported thus far [29][30][31][32][33] are due to a solitary adenoma co-secreting aldosterone and cortisol, as in Cases 1 and 7. However, aldosterone hypersecretion as confirmed by AVS in our 4 patients (Cases 2, 3, 5, 6) was from the contralateral side of the adrenal lesions by imaging side detected by CT imaging in all 8 PA/SCS patients, which is compatible with concomitant cortisol-and/or aldosterone-producing tumors.…”

Section: Discussionmentioning

confidence: 96%

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: Discussionmentioning

confidence: 96%

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

et al. 2011

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“…On the other hand, at least 33 other cases of concurrent autonomous secretion of both aldosterone and cortisol from aldosterone cortisol-producing adenomas (ACPAs) have been reported so far. [6][7][8][9][10][11][12][13][14][15][16][17][18][19][20][21][22] Higher expression of mRNA for 17α-hydroxylase has been found in ACPAs compared to APAs. 22 Furthermore, immunohistochemical analysis of ACPAs has shown that aldosterone synthase on the one hand, and both enzymes specifically involved in cortisol biosynthesis (i.e., 17α-hydroxylase and 11β-hydroxylase) on the other hand, are localized in two different cell types; this finding suggests the existence of two separate cell populations with different steroidogenic pathways in ACPAs.…”

Section: Discussionmentioning

confidence: 99%

Combined Conn's Syndrome and Subclinical Hypercortisolism From an Adrenal Adenoma Associated With Homolateral Renal Carcinoma

Rossi

Foroni

Regolisti

et al. 2008

American Journal of Hypertension

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Primary aldosteronism (PA) is emerging as the most common form of secondary hypertension, with a prevalence of ~5-10% among hypertensive patients. 1,2 The prevalence of aldosterone-producing adenomas (APAs) among patients with PA is ~35-50% in those centers where adrenal venous sampling is expertly performed for the differentiation of APA from idiopathic hyperaldosteronism. Therefore, it is expected that more and more patients with APAs will be detected and surgically cured in the near future. Hence, optimal perioperative management of patients with Conn's syndrome is highly desirable. Postoperative selective hypoaldosteronism with hyperkalemia is a rare, but well-known complication, and monitoring of serum potassium levels after surgery is currently recommended. 2 On the other hand, postoperative hypocortisolism is not currently regarded as a noteworthy complication. Indeed, the coexistence of PA and autonomous hypercortisolism from adrenal adenomas producing both aldosterone and cortisol has been reported anecdotally in western countries. caSe reportA 62-year-old man was referred to our unit on December 2006 for resistant hypertension. He had been discovered with hypertension and hypokalemia at 33 years of age. In the past 10 years, he had been treated with spironolactone 100 mg od, amlodipine 10 mg od, and ramipril 5 mg od. At presentation, physical examination was normal. In particular, no signs of Cushing's syndrome were observed. The patient's blood pressure was 178/102 mm Hg, and body mass index was 23.3 kg/ m 2 . Cardiac ultrasound examination revealed left ventricular hypertrophy (143 g/m 2 ).Laboratory investigations performed 6 weeks after withdrawal of spironolactone showed a combination of PA and adrenocorticotropic hormone (ACTH)-independent hypercortisolism. Indeed, plasma aldosterone was markedly elevated and plasma renin activity relatively suppressed both at baseline and after captopril administration, resulting in a high aldosterone-to-plasma renin activity ratio. Intravenous saline infusion did not suppress plasma aldosterone level. Urinary-free cortisol excretion was threefold the upper limit of the normal range, plasma ACTH at 8:00 AM was suppressed, while plasma cortisol was within the normal range. However, overnight lowdose dexamethasone failed to suppress plasma cortisol level ( Table 1).Computed tomography scan of the abdomen revealed a hypodense mass in the right adrenal, 4.5 × 4.5 × 4.0 cm in diameter, and a 5.6 × 5.1 × 4.8 cm mass at the lower pole of the right kidney (Figure 1).Adrenal venous sampling was not performed, in view of the large size of the right adrenal mass, possibly consistent with a malignant lesion, which indicated right adrenalectomy anyway. Moreover, both the long-lasting (~30 years) hypokalemic hypertension and the very high values of both aldosterone and aldosterone-to-renin ratio were unlikely to result from an occult aldosterone-producing microadenoma in the controlateral, radiologically normal adrenal.Right nephrectomy with right adrenalectomy was performed b...

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“…Previous studies have described abnormalities in glucose homeostasis and metabolism in patients with PA (12,13,14,15,16,17,18,19,20,21,22); however, they often showed inconsistent results due to limitations in sample size, lacking of a matched control group, or nonstandardized diagnosis of PA. Nevertheless, there is growing evidence that aldosterone is involved in the pathogenesis of insulin resistance and MetS (23,24).…”

Section: Introductionmentioning

confidence: 99%

Increased prevalence of diabetes mellitus and the metabolic syndrome in patients with primary aldosteronism of the German Conn's Registry

Hanslik¹,

Wallaschofski²,

Dietz³

et al. 2015

European Journal of Endocrinology

132

105

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Design: Abnormalities in glucose homeostasis have been described in patients with primary aldosteronism (PA) but most studies show inconsistent results. Therefore, we aimed to compare the prevalence of type 2 diabetes mellitus and metabolic syndrome (MetS) in newly diagnosed PA patients to a matched control cohort of the background population. Methods: In total, 305 PA patients of the prospective German Conn's Registry were compared to the population-based Study of Health In Pomerania (SHIP1; nZ2454). A 1:1 match regarding sex, age, and BMI resulted in 269 matched pairs regarding type 2 diabetes and 183 matched pairs regarding MetS. Of the total, 153 PA patients underwent oral glucose tolerance testing (OGTT) at diagnosis and 38 PA patients were reevaluated at follow-up. Results: Type 2 diabetes and MetS were significantly more frequent in PA patients than in the control population (17.2% vs 10.4%, PZ0.03; 56.8% vs 44.8%, PZ0.02 respectively). Also, HbA1c levels were higher in PA patients than in controls (P!0.01). Of the total, 35.3% of non-diabetic PA patients showed an abnormal OGTT ( 1 ⁄ 4 newly diagnosed type 2 diabetes and 3 ⁄ 4 impaired glucose tolerance). PA patients with an abnormal OGTT at baseline presented with significantly improved 2 h OGTT glucose (PZ0.01) at follow-up. We detected a negative correlation between 2 h OGTT glucose levels and serum potassium (P!0.01). Conclusions: Type 2 diabetes and MetS are more prevalent in patients with PA than in controls matched for sex, age, BMI, and blood pressure. This may explain in part the increased cardiovascular disease morbidity and mortality in PA patients.

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Overt Diabetes Mellitus in a Patient with Combined Primary Aldosteronism and Cushing's Syndrome

Abstract: Abstract

Cited by 8 publications

References 17 publications

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

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

Combined Conn's Syndrome and Subclinical Hypercortisolism From an Adrenal Adenoma Associated With Homolateral Renal Carcinoma

Increased prevalence of diabetes mellitus and the metabolic syndrome in patients with primary aldosteronism of the German Conn's Registry

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