Systemic Hormonal Unloading in Unilateral Adrenalectomy in a Patient with Bilateral Adrenal Hyperplasia: A Case Report

Ma. Felisse Carmen Gomez, Florence Rochelle Gan, Erick Mendoza, Leilani B. Mercado Asis

Apr 2019 DOI 10.35460/2546-1621.2018-0055     Access   

Abstract

Background: Unilateral adrenalectomy has not been recommended in the guidelines as a treatment for primary hyperaldosteronism secondary to bilateral adrenal hyperplasia (BAH). Interestingly, recent studies have shown that increased circulation of aldosterone increased oxidative stress, cardiovascular (CV) complications such as atrial fibrillation, myocardial infarction and heart failure; and that unilateral adrenalectomy led to improved CV function. Therefore, recognizing the role of unilateral adrenalectomy in BAH, specifically for improved quality of life is important.

Clinical case: A 47‐year-old hypertensive (highest blood pressure [BP] 150/90 mmHg) woman had a severe headache, muscle weakness, polyuria, and polydipsia. Her serum potassium (K) was low at 3.1 mmol/L (3.5–5 mmol/L). Initial tests showed elevated plasma aldosterone, suppressed plasma renin activity and elevated aldosterone-renin ratio (6.61 ng/dL, <0.1 ng/mL and 66, respectively). Plasma aldosterone after saline suppression test (12.70 ng/dL) confirmed the diagnosis of primary aldosteronism (PA). MRI showed a well-defined, oval-shaped solid nodule in the medial limb of the left adrenal gland (1.8 x 1.2 cm). Bilateral adrenal vein sampling with adrenocorticotropic hormone (ACTH) stimulation test was compatible with BAH (cortisol-corrected aldosterone ratio pre-ACTH stimulation 1.29 and post-ACTH 1.66), with dominant aldosterone secreting left adrenal gland (7200 vs 3760 ng/dL). She was started on spironolactone 200 mg/day and amlodipine 10 mg/day and eventually shifted to eplerenone. Despite the optimal dose of eplerenone and amlodipine, she still experienced severe headaches, palpitations and breakthrough elevations of BP that led to her recurrent admissions. Eplerenone was shifted back to spironolactone (150-200 mg/day) with amlodipine dose (10 mg/day) normalizing her blood pressure and potassium level, yet with persistent headache and muscle weakness. Repeat imaging using CT scan with contrast showed consistent results. Postoperatively, with all medications discontinued the patient was asymptomatic, normotensive (110/70 mmHg) and normokalemic (4.0 mmol/L). One month later, her BP started to increase again at 140/80 mmHg and her K decreased to 3.4 mmol/L. Normalization of said parameters (BP:120/70 mmHg K: 4.1 mmol/L), with stabilization following lower doses of amlodipine (5mg/day) and spironolactone (25 mg/day). Also, all the symptomatology of the patient resolved completely.

Conclusion: This present case exemplifies a unilateral adrenalectomy approach in BAH, which led to improvement in BP and K levels, despite low medication doses. Furthermore, symptom relief and improved quality of life, as desired outcomes, were achieved.

Keywords:

Hormonal unloading, primary aldosteronism, hypertension, bilateral adrenal hyperplasia, hyperaldosteronism, case report

  1. Athyros V, Doumas M, Karagiannis A, Papademetriou V. Arterial Stiffness and Emerging Biomarkers. Angiology. 2015;66(10):901–3.
  2. Kronenberg H, Larsen PR, Melmed S, Polonsky K. Williams Textbook of Endocrinology. Philadelphia, PA: Elsevier; 2015.
  3. Fardella CE, Gordon RD, Loh KC, Mulatero P, Stowasser M, et al. Increased Diagnosis of Primary Aldosteronism, Including Surgically Correctable Forms, in Centers from Five Continents [Internet]. OUP Academic. Oxford University Press; 2004.
  4. Keh-Chuan, Kham, Koay E, Min-Cheh, Shantac E. Prevalence of Primary Aldosteronism among Asian Hypertensive Patients in Singapore 1 [Internet]. OUP Academic. Oxford University Press; 2000.
  5. Fogari R, Preti P, Zoppi A, Rinaldi A, Fogari E, Mugellini A. Prevalence of primary aldosteronism among unselected hypertensive patients: a prospective study based on the use of an aldosterone/renin ratio above 25 as a screening test. [Internet]. Current neurology and neuroscience reports. U.S. National Library of Medicine; 2007 [cited 2018 Dec 28]. Available from: https://www.ncbi.nlm.nih.gov/pubmed/17460380
  6. Hannemann A, Wallaschofski H. Prevalence of primary aldosteronism in patient's cohorts and in population-based studies--a review of the current literature. [Internet]. Current neurology and neuroscience reports. U.S. National Library of Medicine; 2012 [cited 2018 Dec 28]. Available from: https://www.ncbi.nlm.nih.gov/pubmed/22135219
  7. Gumaro M. 8th National Nutrition Survey [Internet]. FNRI Website. [cited 2018 Dec 28]. Available from: http://www.fnri.dost.gov.ph/index.php/nutrition-statistic/19-nutrition-statistic/118-8th-national-nutrition-survey
  8. Omura M, Saito J, Yamaguchi K, Kakuta Y, Nishikawa T. Prospective Study on the Prevalence of Secondary Hypertension among Hypertensive Patients Visiting a General Outpatient Clinic in Japan. Hypertension Research. 2004;27(3):193–202.
  9. Duroin P, Gomez HS, Mercado-Asis LB. Clinical Profile of Patients with Hyperaldosteronism at Santo Tomas University Hospital. Philippine Journal of Internal Medicine. 2002.
  10. Rossi G, Boscaro M, Ronconi V, Funder JW. Aldosterone as a cardiovascular risk factor. Trends in Endocrinology & Metabolism. 2005;16(3):104–7.Catena et al., Aldosterone and the heart: from basic research to clinical evidence. Horm Metab Res. 2012;44:181–187.
  11. Milliez P, Giererd X, Plouin P-F, Blacher J, Safar M, Mourad J-J. Evidence for an Increased Rate of Cardiovascular Events in Patients With Primary Aldosteronism. ACC Current Journal Review. 2005;14(8):18. Savard et al., Cardiovascular Complications Associated With Primary Aldosteronism A Controlled Cross-Sectional Study. Hypertension. 2013;62:331-336.
  12. Matsumura K, Fujii K, Oniki H, Oka M, Iida M. Role of Aldosterone in Left Ventricular Hypertrophy in Hypertension. American Journal of Hypertension. 2006;19(1):13–8. Marney AM and Brown NJ. Aldosterone and end-organ damage. Clin Sci (Lond). 2007;113:267–278.
  13. Strauch B, Petrak O, Wichterle D, Zelinka T, Holaj R, Widimskyjr J. Increased Arterial Wall Stiffness in Primary Aldosteronism in Comparison With Essential Hypertension. American Journal of Hypertension. 2006;19(9):909–14.
  14. Sukor N, Gordon RD, Ku YK, Jones M, Stowasser M. Role of Unilateral Adrenalectomy in Bilateral Primary Aldosteronism: A 22-Year Single Center Experience. The Journal of Clinical Endocrinology & Metabolism. 2009;94(7):2437–45.

Articles related to the one you are viewing

There are currently no results to show, please try again later

CC BY: Open Access Creative Commons Attribution 4.0 International License, which permits use, sharing, adaptation, distribution and reproduction in any medium or format, as long as you give appropriate credit to the original author(s) and the source, provide a link to the Creative Commons license, and indicate if changes were made. The images or other third party material in this article are included in the article’s Creative Commons license, unless indicated otherwise in a credit line to the material. If material is not included in the article’s Creative Commons license and your intended use is not permitted by statutory regulation or exceeds the permitted use, you will need to obtain permission directly from the copyright holder. To view a copy of this license, visit http://creativecommons.org/licenses/by/4.0/