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      A case of primary aldosteronism with secondary hyperparathyroidism and bilateral adrenal tumors

      research-article
      Author(s): ,
      Publication date (Electronic):
      Journal: Endocrinology, Diabetes & Metabolism Case Reports
      Publisher: Bioscientifica Ltd
      Keywords: Adult, Female, Asian - Japanese, Japan, Adrenal, Adrenal, Aldosterone, Mineralocorticoids, PTH, Hyperaldosteronism, Conn's syndrome, Hyperparathyroidism (secondary), Adrenocortical adenoma, Myasthenia, Hypertension, Potassium, Calcium (serum), PTH, TRAP 5b, C1NP, Aldosterone to renin ratio, Aldosterone (24-hour urine), CT scan, Cortisol (serum), ACTH stimulation, Immunohistochemistry, Bone mineral density test, Dexamethasone suppression (low dose), Aldosterone (plasma), Adrenalectomy, Potassium chloride, Radiology/Rheumatology, Surgery, Urology, Unique/unexpected symptoms or presentations of a disease, July, 2015

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          Summary

          A 43-year-old Japanese woman was admitted to our hospital with weakness. Laboratory findings showed hypokalemia, hypocalcemia and elevation of the serum creatinine phosphokinase levels, but intact parathyroid hormone levels. Further evaluations suggested that she had primary aldosteronism (PA), secondary hyperparathyroidism and bilateral adrenal tumors. She was treated successfully by laparoscopic right adrenalectomy. This case not only serves to the diagnosis of bilateral adrenal tumors in which selective adrenal venous sampling (SAVS) proved to be useful, but also for physicians to be aware of secondary hyperparathyroidism and the risk of secondary osteoporosis caused by PA.

          Learning points

          • The classic presenting signs of PA are hypertension and hypokalemia.

          • Hypokalemia can induce rhabdomyolysis.

          • PA causes secondary hyperparathyroidism.

          • Patients with PA have the risk of osteoporosis with secondary hyperparathyroidism.

          • SAVS is useful in bilateral adrenal tumors.

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          Most cited references10

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          Aldosterone and parathyroid hormone interactions as mediators of metabolic and cardiovascular disease.

          Andreas Tomaschitz, Eberhard Ritz, Burkert M. Pieske … (2014)
          Inappropriate aldosterone and parathyroid hormone (PTH) secretion is strongly linked with development and progression of cardiovascular (CV) disease. Accumulating evidence suggests a bidirectional interplay between parathyroid hormone and aldosterone. This interaction may lead to a disproportionally increased risk of CV damage, metabolic and bone diseases. This review focuses on mechanisms underlying the mutual interplay between aldosterone and PTH as well as their potential impact on CV, metabolic and bone health. PTH stimulates aldosterone secretion by increasing the calcium concentration in the cells of the adrenal zona glomerulosa as a result of binding to the PTH/PTH-rP receptor and indirectly by potentiating angiotensin 2 induced effects. This may explain why after parathyroidectomy lower aldosterone levels are seen in parallel with improved cardiovascular outcomes. Aldosterone mediated effects are inappropriately pronounced in conditions such as chronic heart failure, excess dietary salt intake (relative aldosterone excess) and primary aldosteronism. PTH is increased as a result of (1) the MR (mineralocorticoid receptor) mediated calciuretic and magnesiuretic effects with a trend of hypocalcemia and hypomagnesemia; the resulting secondary hyperparathyroidism causes myocardial fibrosis and disturbed bone metabolism; and (2) direct effects of aldosterone on parathyroid cells via binding to the MR. This adverse sequence is interrupted by mineralocorticoid receptor blockade and adrenalectomy. Hyperaldosteronism due to klotho deficiency results in vascular calcification, which can be mitigated by spironolactone treatment. In view of the documented reciprocal interaction between aldosterone and PTH as well as the potentially ensuing target organ damage, studies are needed to evaluate diagnostic and therapeutic strategies to address this increasingly recognized pathophysiological phenomenon.
          Article 0 comments Cited 61 times – based on 0 reviews     Review now
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            Adrenocortical zonation in humans under normal and pathological conditions.

            K Nishimoto, Takeshi Yamazaki, Kuniaki Mukai … (2010)
            Aldosterone synthase (CYP11B2) and steroid 11 beta-hydroxylase (CYP11B1) catalyze the terminal steps for aldosterone and cortisol syntheses, respectively, thereby determining the functional differentiation of human adrenocortical cells. Little is known, however, about how the cells expressing the enzymes are actually distributed in the adrenals under normal and pathological conditions. The objective of the study was to determine the localization of CYP11B2 and -B1 in human adrenal specimens by using developed antibodies capable of distinguishing the two enzymes from each other. Under normal conditions, CYP11B2 was sporadically detected in the zona glomerulosa, whereas CYP11B1 was entirely detected in the zonae fasciculata-reticularis. Adrenocortical cells lacking both enzymes were observed in the outer cortical regions. In addition to conventional zonation, we found a variegated zonation consisting of a subcapsular cell cluster expressing CYP11B2, which we termed aldosterone-producing cell cluster, and a CYP11B1-expressing area. Aldosterone-producing adenomas differed in cell populations expressing CYP11B2 from one another, whereas CYP11B1-expressing and double-negative cells were also intermingled. Adenomas from patients with Cushing's syndrome expressed CYP11B1 entirely but not CYP11B2, resulting in atrophic nontumor glands. The nontumor portions of both types of adenomas bore frequently one or more aldosterone-producing cell clusters, which sustained CYP11B2 expression markedly under the conditions of the suppressed renin-angiotensin system. Immunohistochemistry of the human normal adrenal cortex for CYP11B2 and CYP11B1 revealed a variegated zonation with cell clusters constitutively expressing CYP11B2. This technique may provide a pathological confirmatory diagnosis of adrenocortical adenomas.
            Article 0 comments Cited 47 times – based on 0 reviews     Review now
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              Bone health and aldosterone excess.

              L Ceccoli, V. Ronconi, L Giovannini … (2013)
              A picture of hyperparathyroidism secondary to increased urinary calcium excretion was found in 116 patients with primary aldosteronism (PA), compared with 110 essential hypertensives. After medical or surgical treatment in 40 PA patients, parathyroid hormone (PTH) levels were significantly reduced and bone mineral density (BMD) significantly increased at the lumbar spine, femoral neck, and total hip.
              Article 0 comments Cited 32 times – based on 0 reviews     Review now
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                Author and article information

                Contributors
                Tohru Eguchi
                Journal
                Journal ID (nlm-ta): Endocrinol Diabetes Metab Case Rep
                Journal ID (iso-abbrev): Endocrinol Diabetes Metab Case Rep
                Journal ID (hwp): EDM
                Title: Endocrinology, Diabetes & Metabolism Case Reports
                Publisher: Bioscientifica Ltd (Bristol )
                ISSN (Electronic): 2052-0573
                Publication date (Electronic): 01 August 2015
                Publication date Collection: 01 August 2015
                Volume: 2015
                Electronic Location Identifier: 15-0029
                Affiliations
                [1 ] Department of Internal Medicine , Uwajima City Hospital , 1-1 Gotenmachi, Uwajima, Ehime, 798-8510, Japan
                Article
                Publisher ID: EDM150029
                DOI: 10.1530/EDM-15-0029
                PMC ID: 8142324
                PubMed ID: 30367749
                SO-VID: 2828883b-a68c-4863-b031-66b74a5bb396
                Copyright © © 2015 The authors
                License:

                This work is licensed under a Creative Commons Attribution-NonCommercial-NoDerivs 4.0 Unported License.

                History
                Date received : 28 May 2015
                Date accepted : 19 June 2015
                Categories
                Subject: Adult
                Subject: Female
                Subject: Asian - Japanese
                Subject: Japan
                Subject: Adrenal
                Subject: Adrenal
                Subject: Aldosterone
                Subject: Mineralocorticoids
                Subject: PTH
                Subject: Hyperaldosteronism
                Subject: Conn's Syndrome
                Subject: Hyperparathyroidism (Secondary)
                Subject: Adrenocortical Adenoma
                Subject: Hypertension
                Subject: Potassium
                Subject: Calcium (serum)
                Subject: PTH
                Subject: TRAP 5b
                Subject: C1NP
                Subject: Aldosterone to renin ratio
                Subject: Aldosterone (24-hour urine)
                Subject: CT scan
                Subject: Cortisol (serum)
                Subject: ACTH stimulation
                Subject: Immunohistochemistry
                Subject: Bone mineral density test
                Subject: Dexamethasone suppression (low dose)
                Subject: Aldosterone (plasma)
                Subject: Adrenalectomy
                Subject: Potassium chloride
                Subject: Radiology/Rheumatology
                Subject: Surgery
                Subject: Urology
                Subject: Unique/Unexpected Symptoms or Presentations of a Disease
                Subject: Unique/Unexpected Symptoms or Presentations of a Disease

                Keywords: adult,female,asian - japanese,japan,adrenal,aldosterone,mineralocorticoids,pth,hyperaldosteronism,conn's syndrome,hyperparathyroidism (secondary),adrenocortical adenoma,myasthenia,hypertension,potassium,calcium (serum),trap 5b,c1np,aldosterone to renin ratio,aldosterone (24-hour urine),ct scan,cortisol (serum),acth stimulation,immunohistochemistry,bone mineral density test,dexamethasone suppression (low dose),aldosterone (plasma),adrenalectomy,potassium chloride,radiology/rheumatology,surgery,urology,unique/unexpected symptoms or presentations of a disease,july,2015
                Data availability:
                Keywords: adult, female, asian - japanese, japan, adrenal, aldosterone, mineralocorticoids, pth, hyperaldosteronism, conn's syndrome, hyperparathyroidism (secondary), adrenocortical adenoma, myasthenia, hypertension, potassium, calcium (serum), trap 5b, c1np, aldosterone to renin ratio, aldosterone (24-hour urine), ct scan, cortisol (serum), acth stimulation, immunohistochemistry, bone mineral density test, dexamethasone suppression (low dose), aldosterone (plasma), adrenalectomy, potassium chloride, radiology/rheumatology, surgery, urology, unique/unexpected symptoms or presentations of a disease, july, 2015

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