Primary aldosteronism concurrent with subclinical Cushing's syndrome: a case report and review of the literature

J Med Case Rep. 2020 Feb 20;14(1):32. doi: 10.1186/s13256-020-2353-8.

Abstract

Background: The prevalence of primary aldosteronism concurrent with subclinical Cushing's syndrome was higher than previously thought. Through analyzing a rare clinical case, we summarized the diagnosis and management of primary aldosteronism with subclinical Cushing's syndrome.

Case presentation: A 54-year-old Chinese man of Han nationality was diagnosed as having primary aldosteronism with subclinical Cushing's syndrome. An abdominal computed tomography scan revealed a mass in his left adrenal gland and a mass in his right adrenal gland. After finishing sequential adrenal venous sampling without adrenocorticotropic hormone, the result reminded us that the left and right nodules were responsible for hypercortisolism and aldosterone hypersecretion, respectively. Right and left adrenalectomy were performed successively. The pathological diagnosis was adrenocortical adenoma for both. Histological findings revealed that the right one had positive immunostaining for CYP11B2 and the left one had positive immunostaining for CYP11B1. The immunohistochemistry result helped us to confirm the diagnosis. Somatic KCNJ5 mutation (Leu168Arg) was found in the right tumor; there was no KCNJ5 mutation in the left adrenal tumor.

Conclusions: We suggest that patients with primary aldosteronism should have a low-dose overnight dexamethasone suppression test to screen for hypercortisolism. It can help avoid misdiagnoses and contribute to proper understanding of the adrenal vein sampling result. Making sure of the nidus of aldosterone and cortisol secretion is crucial for the therapy of patients with primary aldosteronism and subclinical Cushing's syndrome.

Keywords: Adrenal vein sampling; Primary aldosteronism; Subclinical Cushing’s syndrome; Subtype.

Publication types

  • Case Reports
  • Review

MeSH terms

  • Adrenal Gland Neoplasms / pathology*
  • Adrenal Glands / blood supply
  • Adrenal Glands / diagnostic imaging
  • Adrenal Glands / surgery
  • Adrenalectomy
  • Adrenocortical Adenoma / pathology*
  • China / epidemiology
  • Cushing Syndrome / complications*
  • Cytochrome P-450 CYP11B2 / analysis
  • G Protein-Coupled Inwardly-Rectifying Potassium Channels / genetics
  • Humans
  • Hyperaldosteronism / complications*
  • Immunohistochemistry
  • Male
  • Middle Aged
  • Steroid 11-beta-Hydroxylase / analysis

Substances

  • G Protein-Coupled Inwardly-Rectifying Potassium Channels
  • KCNJ5 protein, human
  • Cytochrome P-450 CYP11B2
  • Steroid 11-beta-Hydroxylase