Skip to main page content
U.S. flag

An official website of the United States government

Dot gov

The .gov means it’s official.
Federal government websites often end in .gov or .mil. Before sharing sensitive information, make sure you’re on a federal government site.

Https

The site is secure.
The https:// ensures that you are connecting to the official website and that any information you provide is encrypted and transmitted securely.

Access keys NCBI Homepage MyNCBI Homepage Main Content Main Navigation
Case Reports
. 2019 Jun 17;19(1):63.
doi: 10.1186/s12902-019-0395-y.

Hypercortisolism and primary aldosteronism caused by bilateral adrenocortical adenomas: a case report

Kaiyun Ren  1 , Jia Wei  1 , Qilin Liu  1 , Yuchun Zhu  2 , Nianwei Wu  2 , Ying Tang  3 , Qianrui Li  1 , Qianying Zhang  1 , Yerong Yu  1 , Zhenmei An  1 , Jing Chen  4 , Jianwei Li  5
Affiliations
Case Reports

Hypercortisolism and primary aldosteronism caused by bilateral adrenocortical adenomas: a case report

Kaiyun Ren et al. BMC Endocr Disord. .

Abstract

Background: Co-existing Cushing's syndrome and primary aldosteronism caused by bilateral adrenocortical adenomas, secreting cortisol and aldosterone, respectively, have rarely been reported. Precise diagnosis and management of this disorder constitute a challenge to clinicians due to its atypical clinical manifestations and laboratory findings.

Case presentation: We here report a Chinese male patient with co-existing Cushing's syndrome and primary aldosteronism caused by bilateral adrenocortical adenomas, who complained of intermittent muscle weakness for over 3 years. Computed tomography scans revealed bilateral adrenal masses. Undetectable ACTH and unsuppressed cortisol levels by dexamethasone suggested ACTH-independent Cushing's syndrome. Elevated aldosterone to renin ratio and unsuppressed plasma aldosterone concentration after saline infusion test suggested primary aldosteronism. Adrenal venous sampling adjusted by plasma epinephrine revealed hypersecretion of cortisol from the left adrenal mass and of aldosterone from the right one. A sequential bilateral laparoscopic adrenalectomy was performed. The cortisol level was normalized after partial left adrenalectomy and the aldosterone level was normalized after subsequent partial right adrenalectomy. Histopathological evaluation of the resected surgical specimens, including immunohistochemical staining for steroidogenic enzymes, revealed a left cortisol-producing adenoma and a right aldosterone-producing adenoma. The patient's symptoms and laboratory findings resolved after sequential adrenalectomy without any pharmacological treatment.

Conclusions: Adrenal venous sampling is essential in diagnosing bilateral functional adrenocortical adenomas prior to surgery. Proper interpretation of the laboratory findings is particularly important in these patients. Immunohistochemistry may be a valuable tool to identify aldosterone/cortisol-producing lesions and to validate the clinical diagnosis.

Keywords: Adrenal venous sampling; Bilateral adrenal adenomas; Histopathology; Hypercortisolism; Primary aldosteronism.

PubMed Disclaimer

Conflict of interest statement

The authors declare that they have no competing interests.

Figures

Fig. 1
Fig. 1
Adrenal computed tomography (CT) image showing a 19 × 14 mm right adrenal tumor and a 25 × 15 mm left adrenal tumor (arrows)
Fig. 2
Fig. 2
Histological and immunohistochemical findings. (a, c, e, g) Left adrenal gland. (b, d, f, h) Right adrenal gland. (a). Photomicrograph of left adrenal mass showing it is composed mainly of eosinophilic compact cells (H&E stain, × 200). (b). Photomicrograph of right adrenal mass showing it consists of both clear and compact cells, the former type predominating (H&E stain, × 200). (c, d) Immunohistochemical demonstration of 3β-HSD. (e, f) Immunohistochemical demonstration of P450c17. (g, h) Immunohistochemical demonstration of CYP11B2
See this image and copyright information in PMC

References

    1. Xing Y, Lerario AM, Rainey W, Hammer GD. Development of adrenal cortex zonation. Endocrinol Metab Clin N Am. 2015;44(2):243–274. doi: 10.1016/j.ecl.2015.02.001. - DOI - PMC - PubMed
    1. Galati SJ. Primary aldosteronism: challenges in diagnosis and management. Endocrinol Metab Clin N Am. 2015;44(2):355–369. doi: 10.1016/j.ecl.2015.02.010. - DOI - PubMed
    1. Newell-Price J, Bertagna X, Grossman AB, Nieman LK. Cushing's syndrome. Lancet. 2006;367(9522):1605–1617. doi: 10.1016/S0140-6736(06)68699-6. - DOI - PubMed
    1. Späth M, Korovkin S, Antke C, Anlauf M, Willenberg HS. Aldosterone- and cortisol-co-secreting adrenal tumors: the lost subtype of primary aldosteronism. Eur J Endocrinol. 2011;164(4):447–455. doi: 10.1530/EJE-10-1070. - DOI - PubMed
    1. Kukidome D, Miyamura N, Sakakida K, Shimoda S, Shigematu Y, Nishi K, et al. A case of cortisol producing adrenal adenoma associated with a latent aldosteronoma: usefulness of the ACTH loading test for the detection of covert aldosteronism in overt Cushing syndrome. Intern Med. 2012;51(4):395–400. doi: 10.2169/internalmedicine.51.5597. - DOI - PubMed

Publication types