Teragawa H, Oshita C, Orita Y, Hashimoto K, Nakayama H, Yamazaki Y, Sasano H. Primary aldosteronism due to bilateral micronodular hyperplasia and concomitant subclinical Cushing’s syndrome: A case report. World J Clin Cases 2021; 9(5): 1119-1126 [PMID: 33644175 DOI: 10.12998/wjcc.v9.i5.1119]
Corresponding Author of This Article
Hiroki Teragawa, FACC, FACP, FAHA, MD, PhD, Chief Doctor, Department of Cardiovascular Medicine, JR Hiroshima Hospital, 3-1-36 Futabanosato, Higashi-ku, Hiroshima 732-0057, Japan. hiroki-teragawa@jrhh.or.jp
Research Domain of This Article
Endocrinology & Metabolism
Article-Type of This Article
Case Report
Open-Access Policy of This Article
This article is an open-access article which was selected by an in-house editor and fully peer-reviewed by external reviewers. It is distributed in accordance with the Creative Commons Attribution Non Commercial (CC BY-NC 4.0) license, which permits others to distribute, remix, adapt, build upon this work non-commercially, and license their derivative works on different terms, provided the original work is properly cited and the use is non-commercial. See: http://creativecommons.org/licenses/by-nc/4.0/
Hiroki Teragawa, Chikage Oshita, Yuichi Orita, Department of Cardiovascular Medicine, JR Hiroshima Hospital, Hiroshima 732-0057, Japan
Kunihiro Hashimoto, Department of Urology, JR Hiroshima Hospital, Hiroshima 732-0057, Japan
Hirofumi Nakayama, Department of Pathology and Laboratory Medicine, JR Hiroshima Hospital, Hiroshima 732-0057, Japan
Yuto Yamazaki, Hironobu Sasano, Department of Pathology, Tohoku University Graduate School of Medicine, Sendai 980-8574, Japan
Author contributions: Teragawa H, Oshita C and Orita Y performed some endocrinologic and imaging examinations, reviewed the literature and contributed to manuscript drafting; Hashimoto K was the patient’s urological surgeon and contributed to manuscript drafting; Nakayama H performed the histopathological analyses and interpretation and contributed to manuscript drafting; Yamazaki Y and Sasano H performed the histopathological and immunohistochemical analyses and interpretation and contributed to manuscript drafting; all authors issued final approval for the version to be submitted.
Informed consent statement: Informed written consent was obtained from the patient for the surgical procedure, the publication of this report and any accompanying images.
Conflict-of-interest statement: The authors declare that they have no conflict of interest.
CARE Checklist (2016) statement: The authors have read the CARE Checklist (2016), and the manuscript was prepared and revised according to the CARE Checklist (2016).
Open-Access: This article is an open-access article that was selected by an in-house editor and fully peer-reviewed by external reviewers. It is distributed in accordance with the Creative Commons Attribution NonCommercial (CC BY-NC 4.0) license, which permits others to distribute, remix, adapt, build upon this work non-commercially, and license their derivative works on different terms, provided the original work is properly cited and the use is non-commercial. See: http://creativecommons.org/Licenses/by-nc/4.0/
Corresponding author: Hiroki Teragawa, FACC, FACP, FAHA, MD, PhD, Chief Doctor, Department of Cardiovascular Medicine, JR Hiroshima Hospital, 3-1-36 Futabanosato, Higashi-ku, Hiroshima 732-0057, Japan. hiroki-teragawa@jrhh.or.jp
Received: September 10, 2020 Peer-review started: September 10, 2020 First decision: November 23, 2020 Revised: December 3, 2020 Accepted: January 7, 2021 Article in press: January 7, 2021 Published online: February 16, 2021
Abstract
BACKGROUND
Adrenal incidentaloma (AI) has been frequently encountered in the clinical setting. It has been shown that primary aldosteronism (PA) or subclinical Cushing’s syndrome (SCS) are the representative causative diseases of AI. However, the coexistence of PA and SCS has been reportedly observed. Recently, we encountered a case of AI, in which PA and SCS coexisted, confirmed by histopathological examinations after a laparoscopic adrenalectomy. We believe that there were some clinical implications in the diagnosis of the present case.
CASE SUMMARY
A 58-year-old man presented with lower right abdominal pain with a blood pressure of 170/100 mmHg. A subsequent computed tomography scan revealed right ureterolithiasis, which was the cause of right abdominal pain, and right AI measuring 22 mm × 25 mm. After the disappearance of right abdominal pain, subsequent endocrinological examinations were performed. Aldosterone-related evaluations, including adrenal venous sampling, revealed the presence of bilateral PA. In addition, several cortisol-related evaluations showed the presence of SCS on the right adrenal adenoma. A laparoscopic right adrenalectomy was then performed. The histopathological examination of the resected right adrenal revealed the presence of a cortisol-producing adenoma, while CYP11B2 immunoreactivity was absent in this adenoma. However, in the adjacent non-neoplastic adrenal, multiple CYP11B2-positive adrenocortical micronodules were detected, showing the presence of aldosterone-producing adrenocortical micronodules.
CONCLUSION
Careful clinical and pathological examination should be performed when a patient harboring AI presents with concomitant SCS and PA.
Core Tip: Adrenal incidentaloma (AI) has been frequently encountered in the clinical setting. It has been shown that primary aldosteronism (PA) or subclinical Cushing’s syndrome (SCS) are the representative causative diseases of AI. However, the coexistence of PA and SCS has been reportedly observed. In the present case report, we describe a case of AI, in which PA and SCS coexisted after endocrinologic examinations, which was ultimately confirmed by histopathological examinations after a laparoscopic adrenalectomy. Careful clinical examinations should be performed when patients harboring AI present with concomitant SCS and PA.
