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Peer-reviewedCC BY 4.0Frontiers in Endocrinology

Case Report: A case study on bilateral macronodular adrenocortical disease: ACTH-independent hypercortisolism with refractory diabetes and hypertension

  • Yujia Qiu1
  • Pengtao Song2,3
  • Jianping Yao1,4
  • Le Wang5
  • Jiyuan Ying1,4
5 affiliations
  1. 1Department of Endocrinology, Huzhou Central Hospital, Fifth School of Clinical Medicine of Zhejiang Chinese Medical University
  2. 2Department of Pathology, Huzhou Central Hospital, Fifth School of Clinical Medicine of Zhejiang Chinese Medical University
  3. 3Department of Pathology, Huzhou Central Hospital, Affiliated Central Hospital of Huzhou Normal University
  4. 4Department of Endocrinology, Huzhou Central Hospital, Affiliated Central Hospital of Huzhou Normal University
  5. 5Huzhou Central Blood Station
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Abstract

Background: Bilateral macronodular adrenocortical disease is a rare cause of ACTH-independent hypercortisolism. Some patients present without typical Cushingoid features, which may lead to delayed diagnosis.

Case Presentation: A 53-year-old woman presented with poorly controlled diabetes and resistant hypertension despite three antihypertensive agents. She lacked typical Cushingoid features. Laboratory evaluation showed elevated 24-hour urinary free cortisol (UFC) (1656.00 μg/24h; normal: 75.00–520.00 μg/24h), absent circadian rhythm, suppressed ACTH (<5 pg/mL), and non-suppression on low-dose dexamethasone suppression test. Contrast-enhanced CT revealed bilateral adrenal multiple nodular lesions (left: 46×35 mm; right: 57×42 mm). A diagnosis of BMAD with ACTH-independent hypercortisolism without classic Cushingoid features was established.

Management and Outcome: The patient underwent right unilateral adrenalectomy. Postoperatively, glycemic control and blood pressure improved rapidly. At two-year follow-up, cortisol levels remained normal, and both antihypertensive and antidiabetic medications were discontinued without relapse.

Conclusion: BMAD can present with isolated metabolic comorbidities without classic CS features. In selected patients with asymmetric BMAD and ACTH-independent hypercortisolism without classic Cushingoid features, unilateral adrenalectomy of the larger gland may represent a reasonable adrenal-sparing option, achieving sustained biochemical and metabolic remission even in the presence of markedly elevated preoperative UFC levels.

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Cite

BibTeXRIS
BibTeX
@article{qiu2026case,
  title = {{Case Report: A case study on bilateral macronodular adrenocortical disease: ACTH-independent hypercortisolism with refractory diabetes and hypertension}},
  author = {Yujia Qiu and Pengtao Song and Jianping Yao and Le Wang and Jiyuan Ying},
  journal = {Frontiers in Endocrinology},
  year = {2026},
  month = oct,
  doi = {10.3389/fendo.2026.1917551},
  url = {https://www.frontiersin.org/articles/10.3389/fendo.2026.1917551/full},
  note = {CC BY 4.0},
}
RIS
TY  - JOUR
TI  - Case Report: A case study on bilateral macronodular adrenocortical disease: ACTH-independent hypercortisolism with refractory diabetes and hypertension
AU  - Yujia Qiu
AU  - Pengtao Song
AU  - Jianping Yao
AU  - Le Wang
AU  - Jiyuan Ying
PY  - 2026
DA  - 2026/10/19
JO  - Frontiers in Endocrinology
DO  - 10.3389/fendo.2026.1917551
UR  - https://www.frontiersin.org/articles/10.3389/fendo.2026.1917551/full
AB  - Background: Bilateral macronodular adrenocortical disease is a rare cause of ACTH-independent hypercortisolism. Some patients present without typical Cushingoid features, which may lead to delayed diagnosis.  Case Presentation: A 53-year-old woman presented with poorly controlled diabetes and resistant hypertension despite three antihypertensive agents. She lacked typical Cushingoid features. Laboratory evaluation showed elevated 24-hour urinary free cortisol (UFC) (1656.00 μg/24h; normal: 75.00–520.00 μg/24h), absent circadian rhythm, suppressed ACTH (<5 pg/mL), and non-suppression on low-dose dexamethasone suppression test. Contrast-enhanced CT revealed bilateral adrenal multiple nodular lesions (left: 46×35 mm; right: 57×42 mm). A diagnosis of BMAD with ACTH-independent hypercortisolism without classic Cushingoid features was established.  Management and Outcome: The patient underwent right unilateral adrenalectomy. Postoperatively, glycemic control and blood pressure improved rapidly. At two-year follow-up, cortisol levels remained normal, and both antihypertensive and antidiabetic medications were discontinued without relapse.  Conclusion: BMAD can present with isolated metabolic comorbidities without classic CS features. In selected patients with asymmetric BMAD and ACTH-independent hypercortisolism without classic Cushingoid features, unilateral adrenalectomy of the larger gland may represent a reasonable adrenal-sparing option, achieving sustained biochemical and metabolic remission even in the presence of markedly elevated preoperative UFC levels.
SN  - 1664-2392
ER  -