Journal of Yeungnam Medical Science

Case reports

Endocrinology, Diabetes, and Metabolism
Bilateral adrenal adenomas with autonomous cortisol secretion from both glands and autonomous aldosterone secretion from the left adrenal: a case report: Jung Eun Han, Soyeon Yoo, Sang Ah Lee, Gwanpyo Koh; J Yeungnam Med Sci. 2025;42:33. Published online April 28, 2025; DOI: https://doi.org/10.12701/jyms.2025.42.33

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Abstract PDF: Primary aldosteronism (PA) is the most common cause of secondary hypertension and increases the morbidity and mortality associated with cardiovascular diseases. When PA coexists with autonomous cortisol secretion (ACS), the cardiovascular risk increases significantly, especially in cases of bilateral adrenal adenomas with asymmetric hormone secretion, which poses diagnostic and therapeutic challenges. A 50-year-old female presented with hypertension and hypokalemia. PA was diagnosed based on elevated aldosterone levels, suppressed plasma renin activity, and the results of various dynamic endocrine tests. Imaging revealed bilateral adrenal adenomas, and adrenal venous sampling (AVS) confirmed aldosterone hypersecretion from the left adrenal gland and cortisol hypersecretion from both adrenal glands. The patient subsequently underwent left adrenalectomy, which resolved the aldosterone hypersecretion and normalized blood pressure and potassium levels. However, the cortisol hypersecretion persisted. This case highlights the importance of AVS in identifying the sources of hormone secretion and enabling targeted surgical treatment while avoiding bilateral adrenalectomy, which can lead to lifelong adrenal insufficiency. Comprehensive endocrine evaluation, including ACS assessment, in patients with PA is essential to help reduce the cardiovascular risks associated with PA and ACS and thus improve treatment outcomes.

Endocrinology, Diabetes, and Metabolism
Cushing syndrome in pregnancy, diagnosed after delivery: Han Byul Kim, Mi Kyung Kim, El Kim, Keun Soo Ahn, Hye Soon Kim, Nam Kyung Kim; Yeungnam Univ J Med. 2021;38(1):60-64. Published online May 22, 2020; DOI: https://doi.org/10.12701/yujm.2020.00290

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Cushing syndrome (CS) is rare in pregnancy, and few cases have been reported to date. Women with untreated CS rarely become pregnant because of the ovulatory dysfunction induced by hypercortisolism. It is difficult to diagnose CS in pregnancy because of its very low incidence, the overlap between the clinical signs of hypercortisolism and the physiological changes that occur during pregnancy and the changes in hypothalamus-pituitary-adrenal axis activity that occur during pregnancy and limit the value of standard diagnostic testing. However, CS in pregnancy is associated with poor maternal and fetal outcomes; therefore, its early diagnosis and treatment are important. Here, we report two patients with CS that was not diagnosed during pregnancy, in whom maternal and fetal morbidity developed because of hypercortisolism.

Citations

Citations to this article as recorded by

Cushing’s disease with twin pregnancy and diabetes mellitus: a case report and literature review
Hideyasu Asai, Ikuo Yamamori, Shigeru Hagimoto, Kyoichi Okumura, Koki Sakakibara
Endocrine Journal.2024; 71(10): 1003. CrossRef
Endogenous Cushing’s syndrome during pregnancy
Nada Younes, Matthieu St-Jean, Isabelle Bourdeau, André Lacroix
Reviews in Endocrine and Metabolic Disorders.2023; 24(1): 23. CrossRef
Cushing Syndrome in Pregnancy: A Case Presentation and Review of Literature
HamidReza Samimagham, Ava Ziaei, Mohammad Tamaddondar, Mitra Kazemi Jahromi
Journal of Kermanshah University of Medical Sciences.2023;[Epub] CrossRef

Case Report

Endocrinology, Diabetes, and Metabolism
Adrenocorticotropic hormone (ACTH)-producing pheochromocytoma presented as Cushing syndrome and complicated by invasive aspergillosis.: Jae Ho Cho, Da Eun Jeong, Jae Young Lee, Jong Geol Jang, Jun Sung Moon, Mi Jin Kim, Ji Sung Yoon, Kyu Chang Won, Hyoung Woo Lee; Yeungnam Univ J Med. 2015;32(2):132-137. Published online December 31, 2015; DOI: https://doi.org/10.12701/yujm.2015.32.2.132

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Abstract PDF

Adrenocorticotropic hormone (ACTH)-producing pheochromocytoma has been rarely reported, whereas only a few cases of Cushing syndrome accompanied by opportunistic infections have been reported. We experienced a patient with pheochromocytoma with ectopic Cushing syndrome complicated by invasive aspergillosis. A 35-year-old woman presented with typical Cushingoid features. Her basal plasma cortisol, ACTH, and 24-hour urine free cortisol levels were significantly high, and 24-hour urine metanephrine and catecholamine levels were slightly elevated. The endogeneous cortisol secretion was not suppressed by either low- or high-dose dexamethasone. Abdominal computed tomography (CT) revealed a heterogeneous enhancing mass measuring approximately 2.5 cm in size in the left adrenal gland. No definitive mass lesion was observed on sellar magnetic resonance imaging. On fluorine-18 fluorodeoxyglucose positron emission tomography/CT, a hypermetabolic nodule was observed in the left upper lung. Thus, we performed a percutaneous needle biopsy, which revealed inflammation, not malignancy. Thereafter, we performed a laparoscopic left adrenalectomy, and its pathologic finding was a pheochromocytoma with positive immunohistostaining for ACTH. After surgery, the biochemistry was normalized, but the clinical course was fatal despite intensive care because of the invasive aspergillosis that included the lungs, retina, and central nervous system.

Citations

Citations to this article as recorded by

Ectopic ACTH- and/or CRH-Producing Pheochromocytomas
Patrick F Elliott, Thomas Berhane, Oskar Ragnarsson, Henrik Falhammar
The Journal of Clinical Endocrinology & Metabolism.2021; 106(2): 598. CrossRef
Ectopic adrenocorticotrophic hormone syndrome (EAS) with phaeochromocytoma: a challenging endocrine case with a happy ending
Sharifah Faradila Wan Muhamad Hatta, Leoni Lekkakou, Ananth Viswananth, Harit Buch
BMJ Case Reports.2019; 12(8): e230636. CrossRef
Severe Cushing Syndrome Due to an ACTH-Producing Pheochromocytoma: A Case Presentation and Review of the Literature
Jenan N Gabi, Maali M Milhem, Yara E Tovar, Emhemmid S Karem, Alaa Y Gabi, Rodhan A Khthir
Journal of the Endocrine Society.2018; 2(7): 621. CrossRef

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