Journal of Yeungnam Medical Science

Case report

Endocrinology, Diabetes, and Metabolism
Ruptured triple hormone-secreting adrenal cortical carcinoma with hyperaldosteronism, hypercortisolism, and elevated normetanephrine: a case report: Sin Yung Woo, Seongji Park, Kun Young Kwon, Dong-Mee Lim, Keun-Young Park, Jong-Dai Kim; J Yeungnam Med Sci. 2024;41(4):306-311. Published online September 6, 2024; DOI: https://doi.org/10.12701/jyms.2024.00626

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We report a case of a ruptured triple hormone-secreting adrenal mass with hyperaldosteronism, hypercortisolism, and elevated normetanephrine levels, diagnosed as adrenal cortical carcinoma (ACC) by histology. A 53-year-old male patient who initially presented with abdominal pain was referred to our hospital for angiocoagulation of an adrenal mass rupture. Abdominal computed tomography revealed a heterogeneous 19×11×15 cm right adrenal mass with invasion into the right lobe of the liver, inferior vena cava, retrocaval lymph nodes, and aortocaval lymph nodes. Angiocoagulation was performed. Laboratory evaluation revealed excess cortisol via a positive 1-mg overnight dexamethasone suppression test, primary hyperaldosteronism via a positive saline infusion test, and plasma normetanephrine levels three times higher than normal. An adrenal mass biopsy was performed for pathological confirmation to commence palliative chemotherapy because surgical management was not deemed appropriate considering the extent of the tumor. Pathological examination revealed stage T4N1M1 ACC. The patient started the first cycle of adjuvant mitotane therapy along with adjuvant treatment with doxorubicin, cisplatin, and etoposide, and was discharged. Clinical cases of dual cortisol- and aldosterone-secreting ACCs or ACCs presenting as pheochromocytomas have occasionally been reported; however, both are rare. Moreover, to the best of our knowledge, a triple hormone-secreting ACC has not yet been reported. Here, we report a rare case and its management. This case report underscores the necessity of performing comprehensive clinical and biochemical hormone evaluations in patients with adrenal masses because ACC can present with multiple hormone elevations.

Citations

Citations to this article as recorded by

Adrenocortical Carcinoma Presenting With Concurrent Cushing Syndrome and Primary Hyperaldosteronism
Naseem Eisa
AACE Endocrinology and Diabetes.2026;[Epub] CrossRef
Terahertz multiband metamaterial biosensor for ultrasensitive detection of PC-12 adrenal gland carcinoma cells through microenvironmental analysis
Musa N. Hamza, Mohammad Alibakhshikenari, Bal Virdee, Sunil Lavadiya, Iftikhar ud Din, Bruno Sanches, Slawomir Koziel, Syeda Iffat Naqvi, Ali Farmani, Abinash Panda, Hassan Zakeri, Nisar Ahmad Abbasi
Sensors and Actuators A: Physical.2026; 405: 117797. CrossRef

Case Report

Oncology and Cancer Research
Huge pheochromocytoma presented with paraaortic lymph node and spine metastases: Yeon Won Park, Han Ju Moon, Jung Suk Han, Ji Min Han, Jong Wook Park, Yun Hyi Ku; Yeungnam Univ J Med. 2017;34(2):247-253. Published online December 31, 2017; DOI: https://doi.org/10.12701/yujm.2017.34.2.247

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Abstract PDF: Approximately 10–15% of pheochromocytomas are malignant. There are insufficient histologic criteria for the diagnosis of malignant pheochromocytoma. Thus, the term malignant pheochromocytoma is restricted to tumors with local invasion or distant metastases. We experienced a case of malignant pheochromocytoma recurred with spinal metastasis 4 years after the surgery for huge benign pheochromocytoma. A 68-year-old female was admitted for trunk and back pain. The patient had a history of surgery 4 years ago for a 10.0×9.5×7.5 cm sized benign pheochromocytoma at the left adrenal gland. A thoracolumbar magnetic resonance imaging showed a tumor in the 7th thoracic vertebral body and a 24-hour urinary norepinephrine increased, suggesting metastatic recurrence of malignant pheochromocytoma. After metastasectomy in the 7th thoracic vertebral body, urine catecholamine was normalized and pain also disappeared. However, a metastatic lesion was found in the paraaortic area on a follow-up abdominal computed tomography scan and an additional metastasectomy was performed. The pathology confirmed the diagnosis of metastatic pheochromocytoma in the paraaortic lymph nodes. She is supposed to be treated with adjuvant iodine 131-meta-iodobenzylguanidine therapy. In our experience, a close follow-up should be considered in patients who had a huge benign pheochromocytoma due to the possibility of malignant metastases.

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