Endocrinology and Metabolism

Case Report

A Case of Non - Islet Cell Tumor Hypoglycemia.: Moon Hee Lee, Seung Sik Kang, Jin Lee, Sung Hee Ihm, Jae Myung Yoo, Moon Gi Choi, Hyung Joon Yoo, Sung Woo Park; J Korean Endocr Soc. 1995;10(1):65-69. Published online November 6, 2019

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Abstract PDF: The association between non-islet cell tumors and fasting hypoglycemia has been recognized since 1929. The humoral mediator of this non-islet cell tumor hypoglycemia(NICTH) is now known as IGF-II. Hypoglycemia develops commonly in the course of hepatocellular carcinoma(HCC), mostly late in the course and usually mild to moderate in severity. Rarely, severe hypoglycemia occurs early in the course of HCC, suggesting NICTH. We report a case of HCC in which hypoglycemic coma due to NICTH was the presenting symptom.A 52-year-old man developed mental confusion and seizures in the early morning. At emergency room, he was found to have severe hypoglycemia. Circulating levels of insulin, C-peptide, insulinlike growth factor-I(IGF-I) and growth hormone were reduced during hypoglycemia. Diagnostic work-up revealed that he had unresectable HCC. During admission, infusion of more than 400g of dextrose per day in addition to meals was required to prevent hypoglycemia. Because the chemotherapy was refused, he was transferred to a local hospital to receive infusions of dextrose.

Review Article

Diabetes
Latent Autoimmune Diabetes in Adults: Current Status and New Horizons: Paolo Pozzilli, Silvia Pieralice; Endocrinol Metab. 2018;33(2):147-159. Published online June 21, 2018; DOI: https://doi.org/10.3803/EnM.2018.33.2.147

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Abstract PDF PubReader ePub

Autoimmune diabetes is a heterogeneous disease which can arise at any age. Subjects with adult-onset autoimmune diabetes who do not necessitate insulin-therapy for at least 6 months after diagnosis are demarcated as having latent autoimmune diabetes in adults (LADA). This condition is more heterogeneous than young-onset autoimmune diabetes and shares clinical and metabolic characteristics with both type 2 and type 1 diabetes. Patients with LADA are considered by having highly variable β-cell destruction, different degrees of insulin resistance and heterogeneous titre and pattern of islet autoantibody, suggesting different pathophysiological pathways partially explaining the heterogeneous phenotypes of LADA. To date the heterogeneity of LADA does not allow to establish a priori treatment algorithm and no specific guidelines for LADA therapy are available. These subjects are mostly treated as affected by type 2 diabetes, a factor that might lead to the progression to insulin-dependency quickly. A personalised medicine approach is necessary to attain optimal metabolic control and preserve β-cell function to decrease the risk of long-term diabetes complications. Recent data concerning the use of oral antidiabetic agents as dipeptidyl peptidase 4 inhibitors and glucagon-like peptide 1 receptor agonists indicate up-and-coming results in term of protect C-peptide levels and improving glycaemic control. This review summarises current knowledge on LADA, emphasising controversies regarding its pathophysiology and clinical features. Moreover, we discuss data available about novel therapeutic approaches that can be considered for prevention of β-cell loss in LADA.

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Case Reports

A Case of Gastrointestinal Stromal Tumor with Recurrent Hypoglycemia.: Sun Hee Ko, Seok Hwan Kim, Il Ho Maeng, Koon Soon Kim, Yi Sun Jang, Hye Soo Kim, Jong Min Lee, Suk Young Park, Sang Bum Kang; Endocrinol Metab. 2010;25(2):125-130. Published online June 1, 2010; DOI: https://doi.org/10.3803/EnM.2010.25.2.125

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Abstract PDF: Non-islet cell tumor induced hypoglycemia (NICTH) is attributable to overproduction of insulin-like growth factor-II (IGF-II) by solid tumors, and these tumors usually originate from mesenchymal or epithelial cells. Gastrointestinal stromal tumor (GIST) is a rare mesenchymal tumor and most commonly find in the gastrointestinal tract. It is usually expresses the CD117 (stem cell factor receptor, c-kit) detected by immunohistochemistry. Hypoglycemia associated with GIST is very rare and this has not yet been reported in Korea. A 72-year-old man was hospitalized due to frequent episodes of confusion. It was observed that non-hyperinsulinemic hypoglycemia, an elevated serum IGF-II level and a huge liver mass. The histology of liver mass showed c-kit (CD117) positivity, which was consistent with GIST, but it was surgically unresectable. He was treated with imatinib mesylate. Although he recieved palliative treatment, he still experienced intermittent fasting hypoglycemia. After 2 months, the serum IGF-II level was even higher than before. We changed imatinib mesylate to sunitinib malate and performed radiotherapy on the liver mass. Although the change of the liver mass was not significant, he did not suffer from hypoglycemia for three months afterwards.

A Case of Non-islet Cell Tumor Hypoglycemia.: Yun Tae Chae, Il Jun Hwang, Kyung Hee Ryu, Eun Hyang Ko, Jung Im Rue, Soo Kyung Kim, Seok Won Park, Yoo Ri Kim, Yong Wook Cho, Young Kil Choi, Sang Jong Lee; J Korean Endocr Soc. 2006;21(1):74-78. Published online February 1, 2006; DOI: https://doi.org/10.3803/jkes.2006.21.1.74

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Abstract PDF

Mesenchymal tumors including hemangiopericytomas, hepatocellular tumors, adrenal carcinomas, and a variety of other large tumors have been reported to produce excessive amounts of insulin-like growth factor (IGF) type II precursor, which binds weakly to insulin receptors and strongly to IGF-I receptors, leading to insulin like actions. In addition to increased IGF-II production, IGF-II bioavailability is increased due to complex alterations in circulating binding proteins. The authors of this article diagnosed non-islet cell tumor hypoglycemia from an 81-year-old male patient suffering from repetitive fasting hypoglycemia while he has not received any treatment for pulmonary hemangiopericytoma diagnosed in the past. Moreover, this topic is getting reported as the authors have experienced a significant improvement of catamnesis by a treatment with glucocorticoid.

Citations

Citations to this article as recorded by

A Case of Epithelioid Hemangioendothelioma of the Pelvic Retroperitoneum with Hypoglycemia
Ji Ryang Kim, Yun Kyung Jeun, Kee Tae Park, Yang Ho Kang, Seok Man Son, In Ju Kim, Yong Ki Kim, Kyung Un Choi, Kwang Jae Lee
Journal of Korean Endocrine Society.2007; 22(6): 440. CrossRef

A Case of Diabetes Mellitus Caused by Calcitonin and Somatostatin Secreting Pancreatic Islet Tumor.: Jae Hoon Chung, Kwang Won Kim, Byoung Joon Kim, Sung Hoon Kim, Kyung Ah Kim, Myung Sik Lee, Moon Gyu Lee, Yong Ki Min, Jong Ryol Ham, Dong Joon Kim, Hoe Jung Lee, Young Ryoon Oh; J Korean Endocr Soc. 1999;14(2):425-431. Published online January 1, 2001

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Abstract PDF: A case of 39-year-old diabetic patient with a calcitonin and somatostatin secreting pancreatic islet tumor is presented. He had suffered from chronic diarrhea and dyspepsia for 10 years and was diagnosed with diabetes 2 years ago. Abdominal CT revealed a huge abdominal mass which was considered as a neuroendocrine tumor after US-guided needle biopsy. A distal pancreatectomy and splenectomy were performed. Histologically, tumor cells, amanged in solid sheets, showed small nuclei without significant atypia and granular eosinophilic cytoplasm. Tumor cells showed strong immunoreacitivity for calcitonin and somatostatin. The serum clacitonin was markedly elevated (268.7 pmol/L, normal range; 0.9-7.6 pmol/L). After resection of the tumor, diarrhea and dyspepsia diappeared, and oral glucose tolerance test showed normal glucose tolerance with normalization of calcitonin.

A Case of non-islet Cell Tumor Hypoglycemia Due to Gepatoma-increased serum subfraction of big insulin-like growth factor II.: Kwan Woo Lee, Hyun Soo Kim, Yun Suk Chung, Hyun Man Kim, Myung Ho Yoon, Joon Ho Ko, Hyo Chul Kim, Young Soo Kim, Sung Won Cho; J Korean Endocr Soc. 1997;12(4):667-671. Published online January 1, 2001

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Abstract PDF: Hypoglycemia due to non-islet cell tumor is usually associated with hypersecretion of big insulin-like growth factor II (IGF-II). This big IGF-II cannot form ternary IGF complex, and is biologically more active in peripheral tissue, inducing increased glucose utilization and hypoglycemia. A 57-year-old man developed severe hypoglycemia due to hepatocellular carcinoma. To control hypoglycemia, the patient required continuous glucose infusion. The circulating levels of cortisol and free T4 were in the normal range. The plasma levels of insulin, C-peptide, IGF-I, IGF binding protein-3 (IGFBP-3), and total IGF-II levels were decreased. Radioimmunoassay of IGF-II revealed that big IGF-II immunoreactivity markedly increased compared to that of normal control. In this patient, it was strongly suggested that big IGF-II might be a cause of severe intractable hypoglycemia.

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