{"status":"ok","message-type":"work","message-version":"1.0.0","message":{"indexed":{"date-parts":[[2026,7,20]],"date-time":"2026-07-20T14:17:10Z","timestamp":1784557030926,"version":"3.55.0"},"reference-count":13,"publisher":"Bioscientifica","issue":"2","license":[{"start":{"date-parts":[[2023,4,1]],"date-time":"2023-04-01T00:00:00Z","timestamp":1680307200000},"content-version":"unspecified","delay-in-days":0,"URL":"http:\/\/creativecommons.org\/licenses\/by-nc-nd\/4.0\/"}],"content-domain":{"domain":["edm.bioscientifica.com"],"crossmark-restriction":true},"short-container-title":[],"published-print":{"date-parts":[[2023,4,1]]},"abstract":"<jats:sec>\n                    <jats:title>Summary<\/jats:title>\n                    <jats:p>Doege\u2013Potter syndromeis a paraneoplastic syndrome characterized by nonislet cell tumor hypoglycemia due to a solitary fibrous tumor, which produces insulin-like growth factor II. In this report, we present the case of a 67-year-old male with recurrent and refractory hypoglycemia due to DPS successfully treated with imatinib. He initially presented with neuroglycopenic symptoms and dyspnea secondary to a giant tumor in the left hemithorax, which was totally resected. During follow-up, 7 years later, he presented with thoracoabdominal tumor recurrence associated with severe hypoglycemia and underwent subtotal tumor resection, with a subsequent improvement of symptoms. The following year, he had a recurrence of his intra-abdominal tumor, which was unresectable, associated with severe hypoglycemia refractory to dextrose infusion and corticosteroids, thus receiving imatinib with a favorable response. The clinical presentation, diagnostic approach, progression of the disease, and response to treatment with imatinib in the management of a patient with large, recurrent, and unresectable mesenchymal tumors with insulin-like growth factor-2 secretion causing hypoglycemiahighlight the importance of this case report.<\/jats:p>\n                  <\/jats:sec>\n                  <jats:sec>\n                    <jats:title>Learning points<\/jats:title>\n                    <jats:list list-type=\"bullet\">\n                      <jats:list-item>\n                        <jats:p>Doege\u2013Potter syndrome (DPS) is a rare cause of tumoral hypoglycemia of non-pancreatic origin.<\/jats:p>\n                      <\/jats:list-item>\n                      <jats:list-item>\n                        <jats:p>Some malignant or benignant neoplasms have ectopic secretion of insulin-like growth factor-2.<\/jats:p>\n                      <\/jats:list-item>\n                      <jats:list-item>\n                        <jats:p>Total surgical removal is the principal treatment in patients with DPS.<\/jats:p>\n                      <\/jats:list-item>\n                      <jats:list-item>\n                        <jats:p>Tyrosine kinase inhibitors management in DPS may contribute to improved tumor control in patients with unresectable tumors and severe hypoglycemia.<\/jats:p>\n                      <\/jats:list-item>\n                    <\/jats:list>\n                  <\/jats:sec>","DOI":"10.1530\/edm-22-0360","type":"journal-article","created":{"date-parts":[[2023,4,21]],"date-time":"2023-04-21T10:00:13Z","timestamp":1682071213000},"update-policy":"https:\/\/doi.org\/10.1530\/crossmarkpolicy-2","source":"Crossref","is-referenced-by-count":5,"title":["Imatinib use in the management of a patient with Doege\u2013Potter syndrome"],"prefix":"10.1530","volume":"2023","author":[{"ORCID":"https:\/\/orcid.org\/0000-0002-2851-3727","authenticated-orcid":true,"given":"Jose","family":"Paz-Ibarra","sequence":"first","affiliation":[{"name":"Faculty of Medicine, National University of San Marcos, Lima, Peru"},{"name":"National Hospital Edgardo Rebagliati Martins, Lima, Peru"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0000-0003-1645-4640","authenticated-orcid":true,"given":"Jose","family":"Lu-Antara","sequence":"additional","affiliation":[{"name":"Faculty of Medicine, National University of San Marcos, Lima, Peru"},{"name":"Scientific Society of San Fernando, Lima, Peru"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0000-0002-6196-4175","authenticated-orcid":true,"given":"Brenda-Erendida","family":"Uscamayta","sequence":"additional","affiliation":[{"name":"Faculty of Medicine, National University of San Marcos, Lima, Peru"},{"name":"Scientific Society of San Fernando, Lima, Peru"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0000-0001-7103-7730","authenticated-orcid":true,"given":"Jhancy","family":"Martinez-Auris","sequence":"additional","affiliation":[{"name":"Faculty of Medicine, National University of San Marcos, Lima, Peru"},{"name":"Scientific Society of San Fernando, Lima, Peru"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0000-0002-4775-581X","authenticated-orcid":true,"given":"Miriam","family":"Valencia-Rivera","sequence":"additional","affiliation":[{"name":"Faculty of Medicine, National University of San Marcos, Lima, Peru"},{"name":"Scientific Society of San Fernando, Lima, Peru"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0000-0003-2932-380X","authenticated-orcid":true,"given":"Sof\u00eda","family":"S\u00e1enz-Bustamante","sequence":"additional","affiliation":[{"name":"Faculty of Medicine, National University of San Marcos, Lima, Peru"},{"name":"National Hospital Edgardo Rebagliati Martins, Lima, Peru"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0000-0001-8860-0585","authenticated-orcid":true,"given":"Marialejandra","family":"Delgado-Rojas","sequence":"additional","affiliation":[{"name":"Faculty of Medicine, National University of San Marcos, Lima, Peru"},{"name":"National Hospital Edgardo Rebagliati Martins, Lima, Peru"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0000-0003-4061-2602","authenticated-orcid":true,"given":"Julia","family":"Salcedo-Vasquez","sequence":"additional","affiliation":[{"name":"Faculty of Medicine, National University of San Marcos, Lima, Peru"},{"name":"National Hospital Edgardo Rebagliati Martins, Lima, Peru"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0000-0001-9719-1875","authenticated-orcid":true,"given":"Marcio","family":"Concepci\u00f3n-Zavaleta","sequence":"additional","affiliation":[{"name":"Division of Endocrinology. School of Medicine. Norbert Wiener University. Lima, Peru"}],"role":[{"vocabulary":"crossref","role":"author"}]}],"member":"416","reference":[{"key":"bib1","series-title":"Sarcoma","article-title":"Clinical presentation, natural history, and therapeutic approach in patients with solitary fibrous tumor: a retrospective analysis","volume":"2020","author":"Sch\u00f6ffski","year":"2020","unstructured":"Sch\u00f6ffski PTimmermans IHompes DStas MSinnaeve FDe Leyn PCoosemans WVan Raemdonck DHauben ESciot R, Clinical presentation, natural history, and therapeutic approach in patients with solitary fibrous tumor: a retrospective analysis. Sarcoma20202020 1385978. (https:\/\/doi.org\/10.1155\/2020\/1385978)"},{"key":"bib2","series-title":"Cirug\u00eda y Cirujanos","article-title":"Solitary fibrous tumor. Clinic and pathological study of 16 cases","volume":"5","author":"Ceballos","year":"2011","unstructured":"Ceballos MEGDHern\u00e1ndez-Sol\u00eds ACruz-Ortiz HGonz\u00e1lez-Atencio Y & Cicero-Sabido R. Solitary fibrous tumor. Clinic and pathological study of 16 cases. Cirug\u00eda y Cirujanos20115 8."},{"key":"bib3","series-title":"Cureus","article-title":"Doege-Potter syndrome and Pierre-Marie-Bamberger syndrome in a patient with pleural solitary fibrous tumor: a rare case with literature review","volume":"12","author":"Solsi","year":"2020","unstructured":"Solsi APho KShojaie SFindakly D & Noori T. Doege-Potter syndrome and Pierre-Marie-Bamberger syndrome in a patient with pleural solitary fibrous tumor: a rare case with literature review. Cureus202012 e7919. (https:\/\/doi.org\/10.7759\/cureus.7919)"},{"key":"bib4","series-title":"International Journal of Clinical Oncology","first-page":"478","article-title":"The therapeutic challenge of a nonresectable solitary fibrous tumor in a hypoglycemic patient","volume":"11","author":"De Boer","year":"2006","unstructured":"De Boer JJager PLWiggers TNieboer PMachteld Wymenga ANPras EHoogenberg KSleijfer DTSuurmeijer AJH & van der Graaf WT. The therapeutic challenge of a nonresectable solitary fibrous tumor in a hypoglycemic patient. International Journal of Clinical Oncology200611478\u2013481. (https:\/\/doi.org\/10.1007\/s10147-006-0606-1)"},{"key":"bib5","series-title":"Journal of Oncology Pharmacy Practice","first-page":"197","article-title":"Effect of the tyrosine kinase inhibitors (sunitinib, sorafenib, dasatinib, and imatinib) on blood glucose levels in diabetic and nondiabetic patients in general clinical practice","volume":"17","author":"Agostino","year":"2011","unstructured":"Agostino NMChinchilli VMLynch CJKoszyk-Szewczyk AGingrich RSivik J & Drabick JJ. Effect of the tyrosine kinase inhibitors (sunitinib, sorafenib, dasatinib, and imatinib) on blood glucose levels in diabetic and nondiabetic patients in general clinical practice. Journal of Oncology Pharmacy Practice201117197\u2013202. (https:\/\/doi.org\/10.1177\/1078155210378913)"},{"key":"bib6","series-title":"Medicine (Baltimore)","article-title":"Doege\u2013Potter syndrome: A review of the literature including a new case report","volume":"96","author":"Han","year":"2017","unstructured":"Han GZhang ZShen XWang KZhao YHe JGao YShan XXin GLi C, Doege\u2013Potter syndrome: A review of the literature including a new case report. Medicine (Baltimore)201796 e7417. (https:\/\/doi.org\/10.1097\/MD.0000000000007417)"},{"key":"bib7","series-title":"American Journal of Surgical Pathology","first-page":"640","article-title":"Localized benign and malignant fibrous tumors of the pleura. A clinicopathologic review of 223 cases","volume":"13","author":"England","year":"1989","unstructured":"England DMHochholzer L & McCarthy MJ. Localized benign and malignant fibrous tumors of the pleura. A clinicopathologic review of 223 cases. American Journal of Surgical Pathology198913640\u2013658. (https:\/\/doi.org\/10.1097\/00000478-198908000-00003)"},{"key":"bib8","series-title":"Respiratory Research","article-title":"Prognostic value of MIB-1 proliferation index in solitary fibrous tumors of the pleura implemented in a new score \u2013 a multicenter study","volume":"18","author":"Diebold","year":"2017","unstructured":"Diebold MSoltermann AHottinger SHaile SRBubendorf LKomminoth PJochum WGrobholz RTheegarten DBerezowska S, Prognostic value of MIB-1 proliferation index in solitary fibrous tumors of the pleura implemented in a new score \u2013 a multicenter study. Respiratory Research201718 210. (https:\/\/doi.org\/10.1186\/s12931-017-0693-8)"},{"key":"bib9","series-title":"Journal of Cancer Research and Practice","first-page":"68","article-title":"Dacarbazine and bevacizumab improved paraneoplastic Doege\u2013potter syndrome of malignant solitary fibrous tumor","volume":"8","author":"Chen","year":"2021","unstructured":"Chen CTLee JCHuang SC & Chen TW. Dacarbazine and bevacizumab improved paraneoplastic Doege\u2013potter syndrome of malignant solitary fibrous tumor. Journal of Cancer Research and Practice2021868\u201372. (https:\/\/doi.org\/10.4103\/JCRP.JCRP_27_20)"},{"key":"bib10","series-title":"Cancer","first-page":"4939","article-title":"Activity of temozolomide and bevacizumab in the treatment of locally advanced, recurrent, and metastatic hemangiopericytoma and malignant solitary fibrous tumor","volume":"117","author":"Park","year":"2011","unstructured":"Park MSPatel SRLudwig JATrent JCConrad CALazar AJWang WLBoonsirikamchai PChoi HWang X, Activity of temozolomide and bevacizumab in the treatment of locally advanced, recurrent, and metastatic hemangiopericytoma and malignant solitary fibrous tumor. Cancer20111174939\u20134947. (https:\/\/doi.org\/10.1002\/cncr.26098)"},{"key":"bib11","series-title":"Journal of Clinical Endocrinology and Metabolism","first-page":"713","article-title":"Management of non-islet-cell tumor hypoglycemia: a clinical review","volume":"99","author":"Bodnar","year":"2014","unstructured":"Bodnar TWAcevedo MJ & Pietropaolo M. Management of non-islet-cell tumor hypoglycemia: a clinical review. Journal of Clinical Endocrinology and Metabolism201499713\u2013722. (https:\/\/doi.org\/10.1210\/jc.2013-3382)"},{"key":"bib12","series-title":"Lancet. Oncology","first-page":"134","article-title":"Pazopanib for treatment of advanced malignant and dedifferentiated solitary fibrous tumor: a multicentre, single-arm, phase 2 trial","volume":"20","author":"Martin-Broto","year":"2019","unstructured":"Martin-Broto JStacchiotti SLopez-Pousa ARedondo ABernabeu DAlava de ECasali PGItaliano AGutierrez AMoura DS, Pazopanib for treatment of advanced malignant and dedifferentiated solitary fibrous tumor: a multicentre, single-arm, phase 2 trial. Lancet. Oncology201920134\u2013144. (https:\/\/doi.org\/10.1016\/S1470-2045(1830676-4)"},{"key":"bib13","series-title":"Journal of Clinical Oncology","first-page":"1753","article-title":"Correlation of computed tomography and positron emission tomography in patients with metastatic gastrointestinal stromal tumor treated at a single institution with imatinib mesylate: proposal of new computed tomography response criteria","volume":"25","author":"Choi","year":"2007","unstructured":"Choi HCharnsangavej CFaria SCMacapinlac HABurgess MAPatel SRChen LLPodoloff DA & Benjamin RS. Correlation of computed tomography and positron emission tomography in patients with metastatic gastrointestinal stromal tumor treated at a single institution with imatinib mesylate: proposal of new computed tomography response criteria. Journal of Clinical Oncology2007251753\u20131759. 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