Volume 29, Issue 5 (Iran South Med J 2026)
Iran South Med J 2026, 29(5): 363-371 |
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Bajalli Z, Kalantar Hormozi M, Zendehboodi S, Ghanbari Z, Dehghani S. Recurrent Hypoglycemia Resistant to Standard Therapy: A Case Report. Iran South Med J 2026; 29 (5) :363-371
URL: http://ismj.bpums.ac.ir/article-1-2677-en.html
URL: http://ismj.bpums.ac.ir/article-1-2677-en.html
Zohre Bajalli1 
, Mohammadreza Kalantar Hormozi1 , Soudabeh Zendehboodi1 , Zahra Ghanbari1 , Saman Dehghani *2
, Mohammadreza Kalantar Hormozi1 , Soudabeh Zendehboodi1 , Zahra Ghanbari1 , Saman Dehghani *2
1- Department of Internal Medicine, School of Medicine, Bushehr University of Medical Sciences, Bushehr, Iran
2- Department of Internal Medicine, School of Medicine, Bushehr University of Medical Sciences, Bushehr, Iran ,s.dehghani@bpums.ac.ir
2- Department of Internal Medicine, School of Medicine, Bushehr University of Medical Sciences, Bushehr, Iran ,
Abstract: (32 Views)
Background: Non-Islet Cell Tumor Hypoglycemia (NICTH) is a rare paraneoplastic syndrome characterized by the overproduction of insulin-like growth factor-2 (IGF-2) or its precursors by non-pancreatic tumors. Gastrointestinal Stromal Tumors (GISTs) are among the mesenchymal malignancies associated with this condition.
Case Presentation: We report a 63-year-old male with a history of Neurofibromatosis Type I (NF1) and metastatic GIST who presented with recurrent symptomatic hypoglycemia (BS=48 mg/dl). Clinical examination revealed a large abdominal mass and diffuse pedunculated skin lesions. Laboratory investigations during hypoglycemia confirmed non-insulin-mediated hypoglycemia, with suppressed levels of insulin (<0.2 µIU/ml) and C-peptide (<0.01 ng/ml). Concurrent hypercalcemia (Ca=13.8 mg/dl) was also observed. 18F-FDG PET/CT imaging identified a large, hypermetabolic abdominal-pelvic GIST with extensive metastatic involvement.
Results: Based on the diagnosis of NICTH, treatment with daily prednisolone (50 mg) was initiated, successfully maintaining normoglycemia and normal calcium levels. The oncological treatment was transitioned to regorafenib. Despite the management of paraneoplastic symptoms, the patient succumbed to disease progression after a three-year follow-up.
Conclusion: NICTH should be considered a primary differential diagnosis in patients with large mesenchymal tumors presenting with hypoinsulinemic hypoglycemia. Glucocorticoids serve as an effective therapeutic option for stabilizing metabolic derangements in such complex paraneoplastic presentations.
Case Presentation: We report a 63-year-old male with a history of Neurofibromatosis Type I (NF1) and metastatic GIST who presented with recurrent symptomatic hypoglycemia (BS=48 mg/dl). Clinical examination revealed a large abdominal mass and diffuse pedunculated skin lesions. Laboratory investigations during hypoglycemia confirmed non-insulin-mediated hypoglycemia, with suppressed levels of insulin (<0.2 µIU/ml) and C-peptide (<0.01 ng/ml). Concurrent hypercalcemia (Ca=13.8 mg/dl) was also observed. 18F-FDG PET/CT imaging identified a large, hypermetabolic abdominal-pelvic GIST with extensive metastatic involvement.
Results: Based on the diagnosis of NICTH, treatment with daily prednisolone (50 mg) was initiated, successfully maintaining normoglycemia and normal calcium levels. The oncological treatment was transitioned to regorafenib. Despite the management of paraneoplastic symptoms, the patient succumbed to disease progression after a three-year follow-up.
Conclusion: NICTH should be considered a primary differential diagnosis in patients with large mesenchymal tumors presenting with hypoinsulinemic hypoglycemia. Glucocorticoids serve as an effective therapeutic option for stabilizing metabolic derangements in such complex paraneoplastic presentations.
Keywords: Gastrointestinal Stromal Tumor (GIST), Non-Islet Cell Tumor Hypogly-cemia (NICTH), Paraneoplastic Syndrome, IGF-II
Original: Case Report |
Subject:
Endocrine System
Received: 2026/04/16 | Accepted: 2026/08/10 | Published: 2026/10/21
Received: 2026/04/16 | Accepted: 2026/08/10 | Published: 2026/10/21
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