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      A case of renovascular hypertension with incidental primary bilateral macronodular adrenocortical hyperplasia

      research-article
      Author(s): 1 , 1 , 1 , 1 , 2 , 1 , 1 , 3 , 4 , 5 , 5 , 1 , 6 , 1
      Publication date (Electronic):
      Journal: Endocrinology, Diabetes & Metabolism Case Reports
      Publisher: Bioscientifica Ltd
      Keywords: Adult, Male, Asian - Japanese, Japan, Adrenal, Adrenal, Cortisol, Aldosterone, Renin, Hypertension, Macronodular Adrenal Hyperplasia , Diabetes mellitus type 2, Hyperaldosteronism, Hyperosmolar hyperglycaemic state, Hypertension, Collapse, Arteriosclerosis, Renal failure, Arterial stenosis*, Hypercortisolaemia, Dyslipidaemia, Glucose (blood), Blood pressure, Cortisol, CT scan, MRI, Angiography, Immunohistochemistry, Renin plasma activity, Aldosterone (blood), Captopril challenge test*, Adrenal venous sampling, ACTH stimulation, Dexamethasone suppression, Adrenal scintigraphy, PET scan, Histopathology, Creatinine, Urinalysis, Estimated glomerular filtration rate, Insulin tolerance, Haematoxylin and eosin staining, Creatinine (serum), Glucose (blood, fasting), HOMA, Total cholesterol, Triglycerides, Adrenalectomy, Nephrectomy*, Laparoscopic adrenalectomy, Insulin, Doxazosin, Alpha-blockers, Nifedipine, Linagliptin, DPP4 inhibitors, Repaglinide, Meglitinides, Insulin degludec*, Insulin Aspart, Tocopherol*, Atorvastatin, Urology, Novel treatment, August, 2020

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          Abstract

          Summary

          Renovascular hypertension (RVHT) is an important and potentially treatable form of resistant hypertension. Hypercortisolemia could also cause hypertension and diabetes mellitus. We experienced a case wherein adrenalectomy markedly improved blood pressure and plasma glucose levels in a patient with RVHT and low-level autonomous cortisol secretion. A 62-year-old Japanese man had been treated for hypertension and diabetes mellitus for 10 years. He was hospitalized because of a disturbance in consciousness. His blood pressure (BP) was 236/118 mmHg, pulse rate was 132 beats/min, and plasma glucose level was 712 mg/dL. Abdominal CT scanning revealed the presence of bilateral adrenal masses and left atrophic kidney. Abdominal magnetic resonance angiography demonstrated marked stenosis of the left main renal artery. The patient was subsequently diagnosed with atherosclerotic RVHT with left renal artery stenosis. His left adrenal lobular mass was over 40 mm and it was clinically suspected the potential for cortisol overproduction. Therefore, laparoscopic left nephrectomy and adrenalectomy were simultaneously performed, resulting in improved BP and glucose levels. Pathological studies revealed the presence of multiple cortisol-producing adrenal nodules and aldosterone-producing cell clusters in the adjacent left adrenal cortex. In the present case, the activated renin-angiotensin-aldosterone system and cortisol overproduction resulted in severe hypertension, which was managed with simultaneous unilateral nephrectomy and adrenalectomy.

          Learning points:

          • Concomitant activation of the renin-angiotensin-aldosterone system and cortisol overproduction may contribute to the development of severe hypertension and lead to lethal cardiovascular complications.

          • Treatment with simultaneous unilateral nephrectomy and adrenalectomy markedly improves BP and blood glucose levels.

          • CYP11B2 immunohistochemistry staining revealed the existence of aldosterone-producing cell clusters (APCCs) in the adjacent non-nodular adrenal gland, suggesting that APCCs may contribute to aldosterone overproduction in patients with RVHT.

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          Most cited references8

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          Aldosterone-Producing Cell Clusters Frequently Harbor Somatic Mutations and Accumulate With Age in Normal Adrenals

          Kei Omata, Sharath Anand, Daniel H. Hovelson … (2017)
          Context: Aldosterone synthase (CYP11B2) immunohistochemistry and next-generation sequencing (NGS) have revealed the frequent presence of aldosterone-producing cell clusters (APCCs) harboring somatic mutations in aldosterone-regulating genes in adrenals from Americans without defined hypertension status. Objective: Determine the frequency and somatic mutation status of APCCs in a Japanese nonhypertensive cohort. Design, Setting, Patients, and Interventions: Adrenals from 837 consecutive autopsies at a Japanese institution, Tohoku University Hospital, were screened to select 107 unilateral adrenal glands from nonhypertensive patients. APCC score (APCC number/adrenal cortex area per case) was assessed by CYP11B2 immunohistochemistry. DNA from all APCCs and adjacent adrenal cortex was subjected to NGS using two panels targeting aldosterone-regulating genes. Primary Outcome Measure: APCC frequency and somatic mutation spectrum. Results: In 107 adrenals, 61 APCCs were detected (average of 0.6 APCCs per gland). APCC score was positively correlated with age (r = 0.50, P < 0.0001). NGS demonstrated high confidence somatic mutations in 21 of 61 APCCs (34%). Notably, 16 of 21 APCCs (76%) harbored somatic mutations in CACNA1D, the most frequently mutated gene in our previous studies of APCCs in Americans and CYP11B2-positive micronodules in cross-sectional imaging (computed tomography) negative primary aldosteronism (PA), whereas no APCCs harbored mutations in KCNJ5, the most frequently mutated gene in aldosterone-producing adenoma. APCC score was significantly lower than our previous cohort of unilateral computed tomography–negative PA. Conclusions: APCCs are frequent in nonhypertensive Japanese adrenals, accumulate with age, and frequently harbor somatic mutations (most commonly in CACNA1D). The role of APCCs in PA pathobiology and non-PA hypertension warrants further investigation.
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            Unilateral Adrenalectomy as a First-Line Treatment of Cushing's Syndrome in Patients With Primary Bilateral Macronodular Adrenal Hyperplasia

            Emmanuelle Debillon, Sylvie Salenave, Philippe Caron … (2015)
            Article 0 comments Cited 32 times – based on 0 reviews     Review now
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              Adrenal CYP11B1/2 expression in primary aldosteronism: immunohistochemical analysis using novel monoclonal antibodies.

              Yasuhiro Nakamura, Takashi Maekawa, Saulo J.A. Felizola … (2014)
              CYP11B1 and CYP11B2 play pivotal roles in adrenocorticosteroids synthesis. We performed semi-quantitative immunohistochemical analysis of these proteins in adrenals from patients with primary aldosteronism using novel monoclonal antibodies. Clusters of cortical cells positive for CYP11B2 were detected in the zona glomerulosa (ZG) of normal adrenal gland (NA), idiopathic hyperaldosteronism (IHA) and the adjacent adrenal of aldosterone-producing adenoma (APA). In APA, heterogenous immunolocalization of CYP11B2 and diffuse immunoreactivity of CYP11B1 were detected in tumor cells, respectively. The relative immunoreactivity of CYP11B2 in the ZG of adjacent adrenal of APA was significantly lower than that of NA, IHA and APA tumor cells, suggestive of suppressed aldosterone biosynthesis in these cells. These findings did indicate the regulatory mechanisms of aldosterone biosynthesis were different between normal/hyperplastic and neoplastic aldosterone-producing cells in human adrenals. CYP11B2 immunoreactivity in the ZG could also serve as a potential immunohistochemical marker differentiating morphologically hyperplastic ZG of IHA and APA adjacent adrenal.
              Article 0 comments Cited 31 times – based on 0 reviews     Review now
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                Author and article information

                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): 06 August 2020
                Publication date Collection: 2020
                Volume: 2020
                Electronic Location Identifier: 19-0163
                Affiliations
                [1 ]Division of Endocrinology and Hypertension , Department of Cardiovascular and Internal Medicine, Graduate School of Medical Science, Kanazawa University, Kanazawa, Ishikawa, Japan
                [2 ]Department of Internal Medicine , Keiju Medical Center, Nanao, Ishikawa, Japan
                [3 ]Department of Hygiene , Graduate School of Medical Science, Kanazawa University, Kanazawa, Ishikawa, Japan
                [4 ]Health Care Center , Japan Advanced Institute of Science and Technology, Nomi, Ishikawa, Japan
                [5 ]Department of Pathology , Tohoku University Hospital, Sendai, Miyagi, Japan
                [6 ]Department of Health Promotion and Medicine of the Future , Kanazawa University, Kanazawa, Ishikawa, Japan
                Author notes
                Correspondence should be addressed to Y Takeda; Email: takeday@ 123456med.kanazawa-u.ac.jp

                *(T Higashitani and S Karashima contributed equally to this work)

                Author information
                http://orcid.org/0000-0002-5809-2003
                Article
                Publisher ID: EDM190163
                DOI: 10.1530/EDM-19-0163
                PMC ID: 7424347
                SO-VID: e51327e3-485b-4123-bdfd-a04251c45890
                Copyright © © 2020 The authors
                License:

                This work is licensed under a Creative Commons Attribution-NonCommercial-NoDerivatives 4.0 International License..

                History
                Date received : 16 May 2020
                Date accepted : 15 June 2020
                Categories
                Subject: Adult
                Subject: Male
                Subject: Asian - Japanese
                Subject: Japan
                Subject: Adrenal
                Subject: Adrenal
                Subject: Cortisol
                Subject: Aldosterone
                Subject: Renin
                Subject: Hypertension
                Subject: Macronodular Adrenal Hyperplasia
                Subject: Diabetes mellitus type 2
                Subject: Hyperaldosteronism
                Subject: Hyperosmolar hyperglycaemic state
                Subject: Hypertension
                Subject: Collapse
                Subject: Arteriosclerosis
                Subject: Renal failure
                Subject: Arterial stenosis*
                Subject: Hypercortisolaemia
                Subject: Dyslipidaemia
                Subject: Glucose (blood)
                Subject: Blood pressure
                Subject: Cortisol
                Subject: CT scan
                Subject: MRI
                Subject: Angiography
                Subject: Immunohistochemistry
                Subject: Renin plasma activity
                Subject: Aldosterone (blood)
                Subject: Captopril challenge test*
                Subject: Adrenal venous sampling
                Subject: ACTH stimulation
                Subject: Dexamethasone suppression
                Subject: Adrenal scintigraphy
                Subject: PET scan
                Subject: Histopathology
                Subject: Creatinine
                Subject: Urinalysis
                Subject: Estimated glomerular filtration rate
                Subject: Insulin tolerance
                Subject: Haematoxylin and eosin staining
                Subject: Creatinine (serum)
                Subject: Glucose (blood, fasting)
                Subject: HOMA
                Subject: Total cholesterol
                Subject: Triglycerides
                Subject: Adrenalectomy
                Subject: Nephrectomy*
                Subject: Laparoscopic adrenalectomy
                Subject: Insulin
                Subject: Doxazosin
                Subject: Alpha-blockers
                Subject: Nifedipine
                Subject: Linagliptin
                Subject: DPP4 inhibitors
                Subject: Repaglinide
                Subject: Meglitinides
                Subject: Insulin degludec*
                Subject: Insulin Aspart
                Subject: Tocopherol*
                Subject: Atorvastatin
                Subject: Urology
                Subject: Novel Treatment
                Subject: Novel Treatment

                Keywords: adult,male,asian - japanese,japan,adrenal,cortisol,aldosterone,renin,hypertension,macronodular adrenal hyperplasia ,diabetes mellitus type 2,hyperaldosteronism,hyperosmolar hyperglycaemic state,collapse,arteriosclerosis,renal failure,arterial stenosis*,hypercortisolaemia,dyslipidaemia,glucose (blood),blood pressure,ct scan,mri,angiography,immunohistochemistry,renin plasma activity,aldosterone (blood),captopril challenge test*,adrenal venous sampling,acth stimulation,dexamethasone suppression,adrenal scintigraphy,pet scan,histopathology,creatinine,urinalysis,estimated glomerular filtration rate,insulin tolerance,haematoxylin and eosin staining,creatinine (serum),glucose (blood, fasting),homa,total cholesterol,triglycerides,adrenalectomy,nephrectomy*,laparoscopic adrenalectomy,insulin,doxazosin,alpha-blockers,nifedipine,linagliptin,dpp4 inhibitors,repaglinide,meglitinides,insulin degludec*,insulin aspart,tocopherol*,atorvastatin,urology,novel treatment,august,2020
                Data availability:
                Keywords: adult, male, asian - japanese, japan, adrenal, cortisol, aldosterone, renin, hypertension, macronodular adrenal hyperplasia , diabetes mellitus type 2, hyperaldosteronism, hyperosmolar hyperglycaemic state, collapse, arteriosclerosis, renal failure, arterial stenosis*, hypercortisolaemia, dyslipidaemia, glucose (blood), blood pressure, ct scan, mri, angiography, immunohistochemistry, renin plasma activity, aldosterone (blood), captopril challenge test*, adrenal venous sampling, acth stimulation, dexamethasone suppression, adrenal scintigraphy, pet scan, histopathology, creatinine, urinalysis, estimated glomerular filtration rate, insulin tolerance, haematoxylin and eosin staining, creatinine (serum), glucose (blood, fasting), homa, total cholesterol, triglycerides, adrenalectomy, nephrectomy*, laparoscopic adrenalectomy, insulin, doxazosin, alpha-blockers, nifedipine, linagliptin, dpp4 inhibitors, repaglinide, meglitinides, insulin degludec*, insulin aspart, tocopherol*, atorvastatin, urology, novel treatment, august, 2020

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