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Ectopic Adrenocorticotropic Hormone-Producing Metastatic Gastrinoma: A Case Report
Shrikant Tamhane1, Lakshmi P Menon2, Dinesh Edem2
1Endocrinology, Diabetes and Metabolism, Baptist Health, North Little Rock, USA.
Cureus
|October 18, 2023
Summary
Rare pancreatic neuroendocrine tumors secreting both gastrin and adrenocorticotropic hormone (ACTH) present diagnostic challenges. This case highlights successful multidisciplinary treatment for metastatic disease, improving prognosis.
Area of Science:
- Endocrinology
- Oncology
- Gastroenterology
Background:
- Pancreatic neuroendocrine tumors (PNETs) are rare neoplasms with diverse clinical presentations.
- Co-secretion of gastrin and adrenocorticotropic hormone (ACTH) by PNETs is exceptionally uncommon, leading to diagnostic delays.
- Ectopic ACTH syndrome can manifest with severe hypokalemia and Cushing's syndrome.
Observation:
- A patient presented with severe hypokalemia, indicative of ectopic Cushing's syndrome.
- Imaging revealed a pancreatic lesion with liver metastases.
- Biopsy confirmed metastatic neuroendocrine tumor, diagnosed as an ectopic ACTH-producing metastatic gastrinoma.
Findings:
- Elevated 24-hour urinary cortisol levels (9,790 mcg/24 hours) confirmed hypercortisolism.
- Treatment with ketoconazole and somatostatin analogs effectively managed hormonal excess.
- Chemotherapy (capecitabine plus temozolomide) and Y90 radioembolization were employed for metastatic liver disease.
Implications:
- Metastatic PNETs, particularly those with ectopic hormone production, carry a poor prognosis.
- Multidisciplinary therapeutic approaches are crucial for disease control and improved outcomes.
- This case underscores the importance of considering rare hormonal syndromes in complex presentations and highlights effective management strategies.
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