Well-differentiated liposarcoma causing non-islet cell tumor hypoglycemia

Non-islet cell tumor hypoglycemia (NICTH) is a paraneoplastic syndrome that causes severe hypoglycemia. The tumor involved produces high-molecular-weight insulin-like growth factor-II (IGF-II). NICTH can be caused by various benign and malignant tumors. However, NICTH due to liposarcoma (LPS) is rar...

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Vydáno v:	International cancer conference journal Ročník 11; číslo 3; s. 210 - 214
Hlavní autoři:	Igarashi, Yosuke, Hirukawa, Hiroshi, Nakano, Tetsuya, Morimoto, Yuta, Fukuda, Shintaro, Tada, Tetsuya
Médium:	Journal Article
Jazyk:	angličtina
Vydáno:	Singapore Springer Nature Singapore 01.07.2022 Springer Nature B.V
Témata:	Abdomen Antibodies Autoimmune diseases Case Report Case reports Cell differentiation Conflicts of interest Emergency medical care Glucose Hormones Hypoglycemia Insulin Insulin-like growth factor I Insulin-like growth factor II Insulin-like growth factors Laboratories Lipopolysaccharides Liposarcoma Medicine Medicine & Public Health Mesentery Molecular weight Oncology Paraneoplastic syndrome Patients Peptides Small intestine Surgery Surgical Oncology Tomography Tumors Unconsciousness Vomiting Non-islet cell tumor hypoglycemia Well-differentiated liposarcoma Hypoglycemia IGF-II
ISSN:	2192-3183, 2192-3183
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Abstract	Non-islet cell tumor hypoglycemia (NICTH) is a paraneoplastic syndrome that causes severe hypoglycemia. The tumor involved produces high-molecular-weight insulin-like growth factor-II (IGF-II). NICTH can be caused by various benign and malignant tumors. However, NICTH due to liposarcoma (LPS) is rare. A 38-year-old man was brought to the hospital emergency department with complaints of vomiting and unconsciousness. His abdomen was distended, and a large tumor was palpable. He had severe hypoglycemia that required continuous glucose infusion. Radiological examination revealed a large 40-cm mass that filled the abdominal cavity, and LPS of the mesentery was suspected. Laboratory data showed low levels of insulin, C-peptide, and IGF-I concentrations. Subsequently, an LPS-induced NICTH was suspected. Complete resection of the tumor and the encased small intestine was performed. The pathological diagnosis was well-differentiated liposarcoma (WDLPS). The Western immunoblot showed that the high-molecular-weight IGF-II confirmed in the serum preoperatively, had almost disappeared postoperatively. Based on these findings, NICTH caused by a WDLPS was diagnosed. The postoperative course was uneventful. In the last 4 years since the primary operation, the patient had not experienced further hypoglycemia symptoms. Here, we report a case of NICTH caused by a large WDLPS. NICTH should be considered in patients with both severe hypoglycemia and large tumors. In these cases, appropriate treatment including semi-urgent surgery should be provided.
AbstractList	Non-islet cell tumor hypoglycemia (NICTH) is a paraneoplastic syndrome that causes severe hypoglycemia. The tumor involved produces high-molecular-weight insulin-like growth factor-II (IGF-II). NICTH can be caused by various benign and malignant tumors. However, NICTH due to liposarcoma (LPS) is rare. A 38-year-old man was brought to the hospital emergency department with complaints of vomiting and unconsciousness. His abdomen was distended, and a large tumor was palpable. He had severe hypoglycemia that required continuous glucose infusion. Radiological examination revealed a large 40-cm mass that filled the abdominal cavity, and LPS of the mesentery was suspected. Laboratory data showed low levels of insulin, C-peptide, and IGF-I concentrations. Subsequently, an LPS-induced NICTH was suspected. Complete resection of the tumor and the encased small intestine was performed. The pathological diagnosis was well-differentiated liposarcoma (WDLPS). The Western immunoblot showed that the high-molecular-weight IGF-II confirmed in the serum preoperatively, had almost disappeared postoperatively. Based on these findings, NICTH caused by a WDLPS was diagnosed. The postoperative course was uneventful. In the last 4 years since the primary operation, the patient had not experienced further hypoglycemia symptoms. Here, we report a case of NICTH caused by a large WDLPS. NICTH should be considered in patients with both severe hypoglycemia and large tumors. In these cases, appropriate treatment including semi-urgent surgery should be provided.Non-islet cell tumor hypoglycemia (NICTH) is a paraneoplastic syndrome that causes severe hypoglycemia. The tumor involved produces high-molecular-weight insulin-like growth factor-II (IGF-II). NICTH can be caused by various benign and malignant tumors. However, NICTH due to liposarcoma (LPS) is rare. A 38-year-old man was brought to the hospital emergency department with complaints of vomiting and unconsciousness. His abdomen was distended, and a large tumor was palpable. He had severe hypoglycemia that required continuous glucose infusion. Radiological examination revealed a large 40-cm mass that filled the abdominal cavity, and LPS of the mesentery was suspected. Laboratory data showed low levels of insulin, C-peptide, and IGF-I concentrations. Subsequently, an LPS-induced NICTH was suspected. Complete resection of the tumor and the encased small intestine was performed. The pathological diagnosis was well-differentiated liposarcoma (WDLPS). The Western immunoblot showed that the high-molecular-weight IGF-II confirmed in the serum preoperatively, had almost disappeared postoperatively. Based on these findings, NICTH caused by a WDLPS was diagnosed. The postoperative course was uneventful. In the last 4 years since the primary operation, the patient had not experienced further hypoglycemia symptoms. Here, we report a case of NICTH caused by a large WDLPS. NICTH should be considered in patients with both severe hypoglycemia and large tumors. In these cases, appropriate treatment including semi-urgent surgery should be provided. Non-islet cell tumor hypoglycemia (NICTH) is a paraneoplastic syndrome that causes severe hypoglycemia. The tumor involved produces high-molecular-weight insulin-like growth factor-II (IGF-II). NICTH can be caused by various benign and malignant tumors. However, NICTH due to liposarcoma (LPS) is rare. A 38-year-old man was brought to the hospital emergency department with complaints of vomiting and unconsciousness. His abdomen was distended, and a large tumor was palpable. He had severe hypoglycemia that required continuous glucose infusion. Radiological examination revealed a large 40-cm mass that filled the abdominal cavity, and LPS of the mesentery was suspected. Laboratory data showed low levels of insulin, C-peptide, and IGF-I concentrations. Subsequently, an LPS-induced NICTH was suspected. Complete resection of the tumor and the encased small intestine was performed. The pathological diagnosis was well-differentiated liposarcoma (WDLPS). The Western immunoblot showed that the high-molecular-weight IGF-II confirmed in the serum preoperatively, had almost disappeared postoperatively. Based on these findings, NICTH caused by a WDLPS was diagnosed. The postoperative course was uneventful. In the last 4 years since the primary operation, the patient had not experienced further hypoglycemia symptoms. Here, we report a case of NICTH caused by a large WDLPS. NICTH should be considered in patients with both severe hypoglycemia and large tumors. In these cases, appropriate treatment including semi-urgent surgery should be provided. Non-islet cell tumor hypoglycemia (NICTH) is a paraneoplastic syndrome that causes severe hypoglycemia. The tumor involved produces high-molecular-weight insulin-like growth factor-II (IGF-II). NICTH can be caused by various benign and malignant tumors. However, NICTH due to liposarcoma (LPS) is rare. A 38-year-old man was brought to the hospital emergency department with complaints of vomiting and unconsciousness. His abdomen was distended, and a large tumor was palpable. He had severe hypoglycemia that required continuous glucose infusion. Radiological examination revealed a large 40-cm mass that filled the abdominal cavity, and LPS of the mesentery was suspected. Laboratory data showed low levels of insulin, C-peptide, and IGF-I concentrations. Subsequently, an LPS-induced NICTH was suspected. Complete resection of the tumor and the encased small intestine was performed. The pathological diagnosis was well-differentiated liposarcoma (WDLPS). The Western immunoblot showed that the high-molecular-weight IGF-II confirmed in the serum preoperatively, had almost disappeared postoperatively. Based on these findings, NICTH caused by a WDLPS was diagnosed. The postoperative course was uneventful. In the last 4 years since the primary operation, the patient had not experienced further hypoglycemia symptoms. Here, we report a case of NICTH caused by a large WDLPS. NICTH should be considered in patients with both severe hypoglycemia and large tumors. In these cases, appropriate treatment including semi-urgent surgery should be provided.
Author	Tada, Tetsuya Igarashi, Yosuke Hirukawa, Hiroshi Nakano, Tetsuya Morimoto, Yuta Fukuda, Shintaro
Author_xml	– sequence: 1 givenname: Yosuke orcidid: 0000-0003-1998-8074 surname: Igarashi fullname: Igarashi, Yosuke email: igarashi-y@jikei.ac.jp organization: Department of Surgery, Tachikawa General Hospital – sequence: 2 givenname: Hiroshi surname: Hirukawa fullname: Hirukawa, Hiroshi organization: Department of Surgery, Tachikawa General Hospital – sequence: 3 givenname: Tetsuya surname: Nakano fullname: Nakano, Tetsuya organization: Department of Surgery, Tachikawa General Hospital – sequence: 4 givenname: Yuta surname: Morimoto fullname: Morimoto, Yuta organization: Department of Digestive and General Surgery, Kaetsu Hospital – sequence: 5 givenname: Shintaro surname: Fukuda fullname: Fukuda, Shintaro organization: Department of Surgery, Tachikawa General Hospital – sequence: 6 givenname: Tetsuya surname: Tada fullname: Tada, Tetsuya organization: Department of Surgery, Tachikawa General Hospital
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Cites_doi	10.1530/EJE-20-0706 10.1210/jc.2013-3382 10.1186/s12902-020-00624-2 10.1210/er.2012-1033 10.1111/iju.13276 10.1677/ERC-07-0161 10.1016/j.ghir.2006.05.003 10.1210/me.2010-0484 10.2174/1874609812666181205142247 10.3390/cells9102276
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References_xml	– volume: 184 start-page: 41 year: 2021 end-page: 49 ident: CR8 article-title: Diagnostic potential of miR-483 family for IGF-II producing non-islet cell tumor hypoglycemia publication-title: Eur J Endocrinol doi: 10.1530/EJE-20-0706 – volume: 99 start-page: 713 year: 2014 end-page: 722 ident: CR1 article-title: Management of non-islet-cell tumor hypoglycemia: a clinical review publication-title: J Clin Endocrinol Metab doi: 10.1210/jc.2013-3382 – volume: 20 start-page: 148 year: 2020 ident: CR11 article-title: Extraordinarily long-inactive solitary fibrous tumor transformed to produce big insulin-like growth factor-2, leading to hypoglycemia and rapid liposarcoma growth: a case report publication-title: BMC Endocr Disord doi: 10.1186/s12902-020-00624-2 – volume: 34 start-page: 798 year: 2013 end-page: 826 ident: CR4 article-title: Tumors, IGF-2, and hypoglycemia: insights from the clinic, the laboratory, and the historical archive publication-title: Endocr Rev doi: 10.1210/er.2012-1033 – volume: 24 start-page: 162 year: 2017 end-page: 165 ident: CR10 article-title: Retroperitoneal liposarcoma excreting insulin-like growth factor 2 that induced severe hypoglycemia publication-title: Int J Urol doi: 10.1111/iju.13276 – volume: 64 start-page: 185 year: 1965 end-page: 189 ident: CR2 article-title: Hypoglycemia with neoplasia (Doege–Potter syndrome) publication-title: Wis Med J – volume: 14 start-page: 979 year: 2007 end-page: 993 ident: CR3 article-title: Non-islet cell tumour-induced hypoglycaemia: a review of the literature including two new cases publication-title: Endocr Relat Cancer doi: 10.1677/ERC-07-0161 – volume: 16 start-page: 211 year: 2006 end-page: 216 ident: CR9 article-title: Clinical features of insulin-like growth factor-II producing non-islet-cell tumor hypoglycemia publication-title: Growth Horm IGF Res doi: 10.1016/j.ghir.2006.05.003 – volume: 25 start-page: 1456 year: 2011 end-page: 1468 ident: CR5 article-title: Research resource: new and diverse substrates for the insulin receptor isoform A revealed by quantitative proteomics after stimulation with IGF-II or insulin publication-title: Mol Endocrinol doi: 10.1210/me.2010-0484 – volume: 11 start-page: 195 year: 2018 end-page: 200 ident: CR7 article-title: A rare diagnosis after the fall of a 96-year-old woman: Doege–Potter syndrome publication-title: Curr Aging Sci doi: 10.2174/1874609812666181205142247 – volume: 9 start-page: 2276 year: 2020 ident: CR6 article-title: Understanding IGF-II action through insights into receptor binding and activation publication-title: Cells doi: 10.3390/cells9102276 – volume: 25 start-page: 1456 year: 2011 ident: 550_CR5 publication-title: Mol Endocrinol doi: 10.1210/me.2010-0484 – volume: 24 start-page: 162 year: 2017 ident: 550_CR10 publication-title: Int J Urol doi: 10.1111/iju.13276 – volume: 16 start-page: 211 year: 2006 ident: 550_CR9 publication-title: Growth Horm IGF Res doi: 10.1016/j.ghir.2006.05.003 – volume: 9 start-page: 2276 year: 2020 ident: 550_CR6 publication-title: Cells doi: 10.3390/cells9102276 – volume: 11 start-page: 195 year: 2018 ident: 550_CR7 publication-title: Curr Aging Sci doi: 10.2174/1874609812666181205142247 – volume: 20 start-page: 148 year: 2020 ident: 550_CR11 publication-title: BMC Endocr Disord doi: 10.1186/s12902-020-00624-2 – volume: 64 start-page: 185 year: 1965 ident: 550_CR2 publication-title: Wis Med J – volume: 99 start-page: 713 year: 2014 ident: 550_CR1 publication-title: J Clin Endocrinol Metab doi: 10.1210/jc.2013-3382 – volume: 14 start-page: 979 year: 2007 ident: 550_CR3 publication-title: Endocr Relat Cancer doi: 10.1677/ERC-07-0161 – volume: 34 start-page: 798 year: 2013 ident: 550_CR4 publication-title: Endocr Rev doi: 10.1210/er.2012-1033 – volume: 184 start-page: 41 year: 2021 ident: 550_CR8 publication-title: Eur J Endocrinol doi: 10.1530/EJE-20-0706
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SubjectTerms	Abdomen Antibodies Autoimmune diseases Case Report Case reports Cell differentiation Conflicts of interest Emergency medical care Glucose Hormones Hypoglycemia Insulin Insulin-like growth factor I Insulin-like growth factor II Insulin-like growth factors Laboratories Lipopolysaccharides Liposarcoma Medicine Medicine & Public Health Mesentery Molecular weight Oncology Paraneoplastic syndrome Patients Peptides Small intestine Surgery Surgical Oncology Tomography Tumors Unconsciousness Vomiting
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Title	Well-differentiated liposarcoma causing non-islet cell tumor hypoglycemia
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