Paraneoplastic Hypoglycemia Leading to Insulin Independence in a Patient With Type 1 Diabetes
Objective: Non–islet cell tumor hypoglycemia (NICTH) is an uncommon paraneoplastic syndrome associated with mesenchymal neoplasms such as gastrointestinal stromal tumors (GISTs). We report the case of a patient with type 1 diabetes (T1D) and recurrent GIST who not only required discontinuation of in...
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| Format: | Article |
| Language: | English |
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Elsevier
2021-11-01
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| Series: | AACE Clinical Case Reports |
| Subjects: | |
| Online Access: | http://www.sciencedirect.com/science/article/pii/S2376060521000687 |
| _version_ | 1818897040144334848 |
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| author | Nami Safai Haeri, MD Hussain Mahmud, MD Mary T. Korytkowski, MD |
| author_facet | Nami Safai Haeri, MD Hussain Mahmud, MD Mary T. Korytkowski, MD |
| author_sort | Nami Safai Haeri, MD |
| collection | DOAJ |
| description | Objective: Non–islet cell tumor hypoglycemia (NICTH) is an uncommon paraneoplastic syndrome associated with mesenchymal neoplasms such as gastrointestinal stromal tumors (GISTs). We report the case of a patient with type 1 diabetes (T1D) and recurrent GIST who not only required discontinuation of insulin therapy but also required continuous parenteral glucose infusions to prevent hypoglycemia. Methods: A 59-year-old woman with a 24-year history of T1D and recurrent GIST presented with frequent episodes of symptomatic hypoglycemia despite continuous reductions in her insulin therapy. Laboratory workup revealed undetectable insulin and C-peptide, low insulin-like growth factor (IGF) 1, normal IGF-2, and an elevated IGF-2:IGF-1 ratio. Medical management with prednisone alone and, later, in combination with octreotide did not reduce hypoglycemic episodes. Eventually, during hospitalization for severe hypoglycemia, she was treated and discharged with continuous intravenous dextrose infusion. She ultimately required around-the-clock glucose infusions, which helped her maintain what she believed was an acceptable quality of life during her remaining weeks. Discussion: NICTH is characterized by excessive tumor production of IGF-2 or pro-IGF-2, leading to unrestricted glucose uptake in peripheral tissues and hypoglycemia. A diagnosis of NICTH can be made on the basis of low IGF-1 levels in the plasma with normal or elevated IGF-2. Tumor resection is the most definitive treatment for NICTH. Conclusion: This patient with T1D presented with resistant hypoglycemia due to recurrence of an enlarging GIST. She required discontinuation of all insulin therapy and continuous dextrose infusions to maintain euglycemia. |
| first_indexed | 2024-12-19T19:09:51Z |
| format | Article |
| id | doaj.art-d85f91e64724476584c3365f5c892aa4 |
| institution | Directory Open Access Journal |
| issn | 2376-0605 |
| language | English |
| last_indexed | 2024-12-19T19:09:51Z |
| publishDate | 2021-11-01 |
| publisher | Elsevier |
| record_format | Article |
| series | AACE Clinical Case Reports |
| spelling | doaj.art-d85f91e64724476584c3365f5c892aa42022-12-21T20:09:21ZengElsevierAACE Clinical Case Reports2376-06052021-11-0176376378Paraneoplastic Hypoglycemia Leading to Insulin Independence in a Patient With Type 1 DiabetesNami Safai Haeri, MD0Hussain Mahmud, MD1Mary T. Korytkowski, MD2Address correspondence to Dr Nami Safai Haeri, Division of Endocrinology and Metabolism, Department of Medicine, University of Pittsburgh Medical Center, 3601 Fifth Avenue, Suite 3B, Pittsburgh, PA 15213.; Division of Endocrinology and Metabolism, Department of Medicine, University of Pittsburgh Medical Center, Pittsburgh, PennsylvaniaDivision of Endocrinology and Metabolism, Department of Medicine, University of Pittsburgh Medical Center, Pittsburgh, PennsylvaniaDivision of Endocrinology and Metabolism, Department of Medicine, University of Pittsburgh Medical Center, Pittsburgh, PennsylvaniaObjective: Non–islet cell tumor hypoglycemia (NICTH) is an uncommon paraneoplastic syndrome associated with mesenchymal neoplasms such as gastrointestinal stromal tumors (GISTs). We report the case of a patient with type 1 diabetes (T1D) and recurrent GIST who not only required discontinuation of insulin therapy but also required continuous parenteral glucose infusions to prevent hypoglycemia. Methods: A 59-year-old woman with a 24-year history of T1D and recurrent GIST presented with frequent episodes of symptomatic hypoglycemia despite continuous reductions in her insulin therapy. Laboratory workup revealed undetectable insulin and C-peptide, low insulin-like growth factor (IGF) 1, normal IGF-2, and an elevated IGF-2:IGF-1 ratio. Medical management with prednisone alone and, later, in combination with octreotide did not reduce hypoglycemic episodes. Eventually, during hospitalization for severe hypoglycemia, she was treated and discharged with continuous intravenous dextrose infusion. She ultimately required around-the-clock glucose infusions, which helped her maintain what she believed was an acceptable quality of life during her remaining weeks. Discussion: NICTH is characterized by excessive tumor production of IGF-2 or pro-IGF-2, leading to unrestricted glucose uptake in peripheral tissues and hypoglycemia. A diagnosis of NICTH can be made on the basis of low IGF-1 levels in the plasma with normal or elevated IGF-2. Tumor resection is the most definitive treatment for NICTH. Conclusion: This patient with T1D presented with resistant hypoglycemia due to recurrence of an enlarging GIST. She required discontinuation of all insulin therapy and continuous dextrose infusions to maintain euglycemia.http://www.sciencedirect.com/science/article/pii/S2376060521000687gastrointestinal stromal tumorshypoglycemiainsulin-like growth factorsparaneoplastic syndrometype 1 diabetes |
| spellingShingle | Nami Safai Haeri, MD Hussain Mahmud, MD Mary T. Korytkowski, MD Paraneoplastic Hypoglycemia Leading to Insulin Independence in a Patient With Type 1 Diabetes AACE Clinical Case Reports gastrointestinal stromal tumors hypoglycemia insulin-like growth factors paraneoplastic syndrome type 1 diabetes |
| title | Paraneoplastic Hypoglycemia Leading to Insulin Independence in a Patient With Type 1 Diabetes |
| title_full | Paraneoplastic Hypoglycemia Leading to Insulin Independence in a Patient With Type 1 Diabetes |
| title_fullStr | Paraneoplastic Hypoglycemia Leading to Insulin Independence in a Patient With Type 1 Diabetes |
| title_full_unstemmed | Paraneoplastic Hypoglycemia Leading to Insulin Independence in a Patient With Type 1 Diabetes |
| title_short | Paraneoplastic Hypoglycemia Leading to Insulin Independence in a Patient With Type 1 Diabetes |
| title_sort | paraneoplastic hypoglycemia leading to insulin independence in a patient with type 1 diabetes |
| topic | gastrointestinal stromal tumors hypoglycemia insulin-like growth factors paraneoplastic syndrome type 1 diabetes |
| url | http://www.sciencedirect.com/science/article/pii/S2376060521000687 |
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