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Doege-Potter Syndrome; A Case of Solitary Fibrous Pleura Tumor Associated with Severe Hypoglycemia: A Case Report in Internal Medicine

BACKGROUND: Doege-Potter syndrome is a rare paraneoplastic entity that is often diagnosed incidentally during the work-up of hypoglycemia of unclear etiology. It is characterized by a non-islet cell tumor hypoglycemia mostly associated with solitary fibrous tumors. These uncommon tumors have been re...

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Autores principales: Castaldo, Viviana, Domenici, Daniela, Biscosi, Mauro Valentino, Ubiali, Paolo, Miranda, Cesare, Zanette, Giorgio, Mazzon, Cinzia, Tonizzo, Maurizio
Formato: Online Artículo Texto
Lenguaje:English
Publicado: Bentham Science Publishers 2023
Materias:
Acceso en línea:https://www.ncbi.nlm.nih.gov/pmc/articles/PMC10548549/
https://www.ncbi.nlm.nih.gov/pubmed/37357513
http://dx.doi.org/10.2174/1871530323666230623112047
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author Castaldo, Viviana
Domenici, Daniela
Biscosi, Mauro Valentino
Ubiali, Paolo
Miranda, Cesare
Zanette, Giorgio
Mazzon, Cinzia
Tonizzo, Maurizio
author_facet Castaldo, Viviana
Domenici, Daniela
Biscosi, Mauro Valentino
Ubiali, Paolo
Miranda, Cesare
Zanette, Giorgio
Mazzon, Cinzia
Tonizzo, Maurizio
author_sort Castaldo, Viviana
collection PubMed
description BACKGROUND: Doege-Potter syndrome is a rare paraneoplastic entity that is often diagnosed incidentally during the work-up of hypoglycemia of unclear etiology. It is characterized by a non-islet cell tumor hypoglycemia mostly associated with solitary fibrous tumors. These uncommon tumors have been reported in <5% of solitary fibrous tumors. Although not unique in its kind, this case is extremely important as this syndrome often conceals unrecognized tumors that can be surgically resolved. CASE PRESENTATION: We present the case of a 59-year-old non-diabetic man with a 2-month history of severe and recurrent fasting hypoglycaemia presenting with severe dyspnea and sweating. Further work-up revealed low insulin, C-peptide, and IGF-1 levels and a large right in-trathoracic solitary fibrous tumor. Unfortunately, bioassays for IGF-2 were unavailable at our hos-pital. Nevertheless, as hypoglycemia completely resolved after resection of the mass, Doege-Potter syndrome was highly suspected. CONCLUSION: Doege-Potter syndrome is a complication of rare tumors. If hy-poglycemia is unexplained, this syndrome should always be suspected, and the presence of un-known masses should be investigated.
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spelling pubmed-105485492023-10-05 Doege-Potter Syndrome; A Case of Solitary Fibrous Pleura Tumor Associated with Severe Hypoglycemia: A Case Report in Internal Medicine Castaldo, Viviana Domenici, Daniela Biscosi, Mauro Valentino Ubiali, Paolo Miranda, Cesare Zanette, Giorgio Mazzon, Cinzia Tonizzo, Maurizio Endocr Metab Immune Disord Drug Targets Pharmacology, Medicine, Endocrinology, Immunology, Inflammation and Allergy, Biochemistry and Molecular Biology BACKGROUND: Doege-Potter syndrome is a rare paraneoplastic entity that is often diagnosed incidentally during the work-up of hypoglycemia of unclear etiology. It is characterized by a non-islet cell tumor hypoglycemia mostly associated with solitary fibrous tumors. These uncommon tumors have been reported in <5% of solitary fibrous tumors. Although not unique in its kind, this case is extremely important as this syndrome often conceals unrecognized tumors that can be surgically resolved. CASE PRESENTATION: We present the case of a 59-year-old non-diabetic man with a 2-month history of severe and recurrent fasting hypoglycaemia presenting with severe dyspnea and sweating. Further work-up revealed low insulin, C-peptide, and IGF-1 levels and a large right in-trathoracic solitary fibrous tumor. Unfortunately, bioassays for IGF-2 were unavailable at our hos-pital. Nevertheless, as hypoglycemia completely resolved after resection of the mass, Doege-Potter syndrome was highly suspected. CONCLUSION: Doege-Potter syndrome is a complication of rare tumors. If hy-poglycemia is unexplained, this syndrome should always be suspected, and the presence of un-known masses should be investigated. Bentham Science Publishers 2023-09-15 2023-09-15 /pmc/articles/PMC10548549/ /pubmed/37357513 http://dx.doi.org/10.2174/1871530323666230623112047 Text en © 2023 Bentham Science Publishers https://creativecommons.org/licenses/by/4.0/© 2023 The Author(s). Published by Bentham Science Publisher. This is an open access article published under CC BY 4.0 https://creativecommons.org/licenses/by/4.0/legalcode)
spellingShingle Pharmacology, Medicine, Endocrinology, Immunology, Inflammation and Allergy, Biochemistry and Molecular Biology
Castaldo, Viviana
Domenici, Daniela
Biscosi, Mauro Valentino
Ubiali, Paolo
Miranda, Cesare
Zanette, Giorgio
Mazzon, Cinzia
Tonizzo, Maurizio
Doege-Potter Syndrome; A Case of Solitary Fibrous Pleura Tumor Associated with Severe Hypoglycemia: A Case Report in Internal Medicine
title Doege-Potter Syndrome; A Case of Solitary Fibrous Pleura Tumor Associated with Severe Hypoglycemia: A Case Report in Internal Medicine
title_full Doege-Potter Syndrome; A Case of Solitary Fibrous Pleura Tumor Associated with Severe Hypoglycemia: A Case Report in Internal Medicine
title_fullStr Doege-Potter Syndrome; A Case of Solitary Fibrous Pleura Tumor Associated with Severe Hypoglycemia: A Case Report in Internal Medicine
title_full_unstemmed Doege-Potter Syndrome; A Case of Solitary Fibrous Pleura Tumor Associated with Severe Hypoglycemia: A Case Report in Internal Medicine
title_short Doege-Potter Syndrome; A Case of Solitary Fibrous Pleura Tumor Associated with Severe Hypoglycemia: A Case Report in Internal Medicine
title_sort doege-potter syndrome; a case of solitary fibrous pleura tumor associated with severe hypoglycemia: a case report in internal medicine
topic Pharmacology, Medicine, Endocrinology, Immunology, Inflammation and Allergy, Biochemistry and Molecular Biology
url https://www.ncbi.nlm.nih.gov/pmc/articles/PMC10548549/
https://www.ncbi.nlm.nih.gov/pubmed/37357513
http://dx.doi.org/10.2174/1871530323666230623112047
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