{"status":"ok","message-type":"work","message-version":"1.0.0","message":{"indexed":{"date-parts":[[2026,7,20]],"date-time":"2026-07-20T14:17:29Z","timestamp":1784557049795,"version":"3.55.0"},"reference-count":24,"publisher":"Bioscientifica","content-domain":{"domain":["edm.bioscientifica.com"],"crossmark-restriction":true},"short-container-title":[],"published-print":{"date-parts":[[2015,10,1]]},"abstract":"Summary<\/jats:title>\n We report the case of a 70-year-old previously healthy female who presented acutely to the Accident and Emergency department with left-sided vasomotor symptoms including reduced muscle tone, weakness upon walking and slurred speech. Physical examination confirmed hemiparesis with VIIth nerve palsy and profound hepatomegaly. A random glucose was low at 1.7\u200ammol\/l, which upon correction resolved her symptoms. In hindsight, the patient recalled having had similar episodes periodically over the past 3 months to which she did not give much attention. While hospitalized, she continued having episodes of symptomatic hypoglycaemia during most nights, requiring treatment with i.v. dextrose and\/or glucagon. Blood tests including insulin and C-peptide were invariably suppressed, in correlation with low glucose. A Synacthen stimulation test was normal (Cort (0\u2032) 390\u200anmol\/l, Cort (30\u2032) 773\u200anmol\/l). A computed tomography scan showed multiple lobulated masses in the abdomen, liver and pelvis. An ultrasound guided biopsy of one of the pelvic masses was performed. Immunohistochemistry supported the diagnosis of a gastrointestinal stromal tumour (GIST) positive for CD34 and CD117. A diagnosis of a non islet cell tumour hypoglycaemia (NICTH) secondary to an IGF2 secreting GIST was confirmed with further biochemical investigations (IGF2=96.5\u200anmol\/l; IGF2:IGF1 ratio 18.9, ULN <10). Treatment with growth hormone resolved the patient's hypoglycaemic symptoms and subsequent targeted therapy with Imatinib was successful in controlling disease progression over an 8-year observation period.<\/jats:p>\n \n Learning points<\/jats:title>\n \n \n \n NICTH can be a rare complication of GISTs that may manifest with severe hypoglycaemia and neuroglucopenic symptoms.<\/jats:p>\n <\/jats:list-item>\n \n NICTH can masquerade as other pathologies thus causing diagnostic confusion.<\/jats:p>\n <\/jats:list-item>\n \n Histological confirmation of GIST induced NICTH and exclusion of other conditions causing hypoglycaemia is essential.<\/jats:p>\n <\/jats:list-item>\n \n Mutational analysis of GISTs should be carried out in all cases as it guides treatment decision.<\/jats:p>\n <\/jats:list-item>\n \n Tailored management of hypoglycaemia, in this case using growth hormone and targeted cyto-reductive therapy, minimizes the risk of possible life-threatening complications.<\/jats:p>\n <\/jats:list-item>\n <\/jats:list>\n <\/jats:p>\n <\/jats:sec>","DOI":"10.1530\/edm-15-0062","type":"journal-article","created":{"date-parts":[[2015,10,23]],"date-time":"2015-10-23T10:37:58Z","timestamp":1445596678000},"update-policy":"https:\/\/doi.org\/10.1530\/crossmarkpolicy-2","source":"Crossref","is-referenced-by-count":1,"title":["Severe paraneoplastic hypoglycemia secondary to a gastrointestinal stromal tumour masquerading as a stroke"],"prefix":"10.1530","volume":"2015","author":[{"given":"G K","family":"Dimitriadis","sequence":"first","affiliation":[{"name":"1 Warwick Institute for the Study of Endocrinology Diabetes and Metabolism (WISDEM Centre), The Arden NET Centre, University Hospitals of Coventry and Warwickshire, UHCW NHS Trust, ENETS CoE, Coventry, UK"},{"name":"2 Division of Experimental Medicine, Faculty of Medicine, Imperial College London, Hammersmith Campus, London, UK"},{"name":"3 Division of Translational and Systems Medicine, Warwick Medical School, University of Warwick, Coventry, UK"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"K","family":"Gopalakrishnan","sequence":"additional","affiliation":[{"name":"5 Department of Histopathology, Coventry and Warwickshire, Pathology Service, UHCW NHS Trust, Coventry, UK"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"R","family":"Rao","sequence":"additional","affiliation":[{"name":"1 Warwick Institute for the Study of Endocrinology Diabetes and Metabolism (WISDEM Centre), The Arden NET Centre, University Hospitals of Coventry and Warwickshire, UHCW NHS Trust, ENETS CoE, Coventry, UK"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"D K","family":"Grammatopoulos","sequence":"additional","affiliation":[{"name":"3 Division of Translational and Systems Medicine, Warwick Medical School, University of Warwick, Coventry, UK"},{"name":"4 Department of Clinical Biochemistry and Histopathology, Coventry and Warwickshire, Pathology Service, UHCW NHS Trust, Coventry, UK"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"H S","family":"Randeva","sequence":"additional","affiliation":[{"name":"1 Warwick Institute for the Study of Endocrinology Diabetes and Metabolism (WISDEM Centre), The Arden NET Centre, University Hospitals of Coventry and Warwickshire, UHCW NHS Trust, ENETS CoE, Coventry, UK"},{"name":"3 Division of Translational and Systems Medicine, Warwick Medical School, University of Warwick, Coventry, UK"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"M O","family":"Weickert","sequence":"additional","affiliation":[{"name":"1 Warwick Institute for the Study of Endocrinology Diabetes and Metabolism (WISDEM Centre), The Arden NET Centre, University Hospitals of Coventry and Warwickshire, UHCW NHS Trust, ENETS CoE, Coventry, UK"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"N","family":"Murthy","sequence":"additional","affiliation":[{"name":"1 Warwick Institute for the Study of Endocrinology Diabetes and Metabolism (WISDEM Centre), The Arden NET Centre, University Hospitals of Coventry and Warwickshire, UHCW NHS Trust, ENETS CoE, Coventry, UK"}],"role":[{"vocabulary":"crossref","role":"author"}]}],"member":"416","reference":[{"key":"ref01","doi-asserted-by":"crossref","first-page":"1582","DOI":"10.1093\/annonc\/mdp038","article-title":"Insulin-like growth factors and insulin-like growth factor-binding proteins in relation to disease status and incidence of hypoglycaemia in patients with a gastrointestinal stromal tumour","volume":"20","year":"2009","journal-title":"Annals of Oncology"},{"key":"ref11","doi-asserted-by":"publisher","first-page":"21","DOI":"10.1093\/annonc\/mdu255)","article-title":"Sarcoma Guidelines of Supplement iii iii","volume":"25","author":"ESMO","year":"2014","journal-title":"Annals Oncology"},{"key":"ref211","doi-asserted-by":"crossref","first-page":"402","DOI":"10.1046\/j.1365-2265.2003.01848.x","article-title":"Hypoglycaemia in a patient with a gastrointestinal stromal tumour","volume":"59","year":"2003","journal-title":"Clinical Endocrinology"},{"key":"ref61","doi-asserted-by":"crossref","first-page":"13","DOI":"10.1186\/1471-2407-7-13","article-title":"Severe paraneoplastic hypoglycaemia in a patient with a gastrointestinal stromal tumour with an exon 9 mutation: a case report","volume":"7","year":"2007","journal-title":"BMC Cancer"},{"key":"ref81","doi-asserted-by":"crossref","first-page":"91","DOI":"10.1016\/S0889-8529(18)30390-6","article-title":"Hypoglycemia in patients with non-islet cell tumors","volume":"18","year":"1989","journal-title":"Endocrinology and Metabolism Clinics of North America"},{"key":"ref121","doi-asserted-by":"crossref","first-page":"1582","DOI":"10.1093\/annonc\/mdp038","article-title":"Insulin-like growth factors and insulin-like growth factor-binding proteins in relation to disease status and incidence of hypoglycaemia in patients with a gastrointestinal stromal tumour","volume":"20","year":"2009","journal-title":"Annals of Oncology"},{"key":"ref31","doi-asserted-by":"crossref","first-page":"457","DOI":"10.1111\/j.1365-2265.2004.01989.x","article-title":"The effectiveness of different treatment options for non-islet cell tumour hypoglycaemia","volume":"60","year":"2004","journal-title":"Clinical Endocrinology"},{"key":"ref71","doi-asserted-by":"crossref","first-page":"711","DOI":"10.1016\/S0025-6196(12)62083-7","article-title":"Functioning insulinoma\u2013incidence, recurrence, and long-term survival of patients: a 60-year study","volume":"66","year":"1991","journal-title":"Mayo Clinic Proceedings"},{"key":"ref221","doi-asserted-by":"publisher","first-page":"24","DOI":"10.1200\/JCO.2006.06.5318","article-title":"JongFABoonstraJGvan Non - islet - cell tumor induced hypoglycemia in patients with advanced gastrointestinal stromal tumor possibly worsened by imatinib of","author":"HambergPde","year":"2006","journal-title":"Journal Clinical Oncology"},{"key":"ref91","doi-asserted-by":"crossref","first-page":"402","DOI":"10.1046\/j.1365-2265.2003.01848.x","article-title":"Hypoglycaemia in a patient with a gastrointestinal stromal tumour","volume":"59","year":"2003","journal-title":"Clinical Endocrinology"},{"key":"ref41","doi-asserted-by":"crossref","first-page":"99","DOI":"10.1016\/S0003-4266(04)95641-7","article-title":"Non-islet cell hypoglycemia","volume":"65","year":"2004","journal-title":"Annales d'Endocrinologie"},{"key":"ref201","doi-asserted-by":"crossref","first-page":"91","DOI":"10.1016\/S0889-8529(18)30390-6","article-title":"Hypoglycemia in patients with non-islet cell tumors","volume":"18","year":"1989","journal-title":"Endocrinology and Metabolism Clinics of North America"},{"key":"ref51","doi-asserted-by":"crossref","first-page":"246","DOI":"10.1016\/j.clinthera.2005.02.004","article-title":"Treatment of hypoglycemia using combined glucocorticoid and recombinant human growth hormone in a patient with a metastatic non-islet cell tumor hypoglycemia","volume":"27","year":"2005","journal-title":"Clinical Therapeutics"},{"key":"ref231","doi-asserted-by":"crossref","first-page":"6809","DOI":"10.1200\/JCO.2005.02.4828","article-title":"Report of two cases severe hypoglycemia caused by paraneoplastic production of IGF-II in patients with advanced gastrointestinal stromal tumours","volume":"23","year":"2005","journal-title":"Journal of Clinical Oncology"},{"key":"ref151","doi-asserted-by":"crossref","first-page":"457","DOI":"10.1111\/j.1365-2265.2004.01989.x","article-title":"The effectiveness of different treatment options for non-islet cell tumour hypoglycaemia","volume":"60","year":"2004","journal-title":"Clinical Endocrinology"},{"key":"ref101","doi-asserted-by":"publisher","first-page":"24","DOI":"10.1200\/JCO.2006.06.5318","article-title":"JongFABoonstraJGvan Non - islet - cell tumor induced hypoglycemia in patients with advanced gastrointestinal stromal tumor possibly worsened by imatinib of","author":"HambergPde","year":"2006","journal-title":"Journal Clinical Oncology"},{"key":"ref191","doi-asserted-by":"crossref","first-page":"711","DOI":"10.1016\/S0025-6196(12)62083-7","article-title":"Functioning insulinoma\u2013incidence, recurrence, and long-term survival of patients: a 60-year study","volume":"66","year":"1991","journal-title":"Mayo Clinic Proceedings"},{"key":"ref131","doi-asserted-by":"publisher","first-page":"21","DOI":"10.1093\/annonc\/mdu255)","article-title":"Sarcoma Guidelines of Supplement iii iii","volume":"25","author":"ESMO","year":"2014","journal-title":"Annals Oncology"},{"key":"ref161","doi-asserted-by":"crossref","first-page":"99","DOI":"10.1016\/S0003-4266(04)95641-7","article-title":"Non-islet cell hypoglycemia","volume":"65","year":"2004","journal-title":"Annales d'Endocrinologie"},{"key":"ref21","first-page":"221","article-title":"Uncontrolled insulin secretion from a childhood pancreatic beta-cell adenoma is not due to the functional loss of ATP sensitive potassium channels","volume":"9","year":"2002","journal-title":"Endocrine-Related Cancer"},{"key":"ref141","first-page":"221","article-title":"Uncontrolled insulin secretion from a childhood pancreatic beta-cell adenoma is not due to the functional loss of ATP sensitive potassium channels","volume":"9","year":"2002","journal-title":"Endocrine-Related Cancer"},{"key":"ref181","doi-asserted-by":"crossref","first-page":"13","DOI":"10.1186\/1471-2407-7-13","article-title":"Severe paraneoplastic hypoglycaemia in a patient with a gastrointestinal stromal tumour with an exon 9 mutation: a case report","volume":"7","year":"2007","journal-title":"BMC Cancer"},{"key":"ref111","doi-asserted-by":"crossref","first-page":"6809","DOI":"10.1200\/JCO.2005.02.4828","article-title":"Report of two cases severe hypoglycemia caused by paraneoplastic production of IGF-II in patients with advanced gastrointestinal stromal tumours","volume":"23","year":"2005","journal-title":"Journal of Clinical Oncology"},{"key":"ref171","doi-asserted-by":"crossref","first-page":"246","DOI":"10.1016\/j.clinthera.2005.02.004","article-title":"Treatment of hypoglycemia using combined glucocorticoid and recombinant human growth hormone in a patient with a metastatic non-islet cell tumor hypoglycemia","volume":"27","year":"2005","journal-title":"Clinical Therapeutics"}],"container-title":["Endocrinology, Diabetes & Metabolism Case Reports"],"original-title":[],"link":[{"URL":"https:\/\/edm.bioscientifica.com\/view\/journals\/edm\/2015\/1\/EDM15-0062.xml","content-type":"text\/html","content-version":"vor","intended-application":"text-mining"},{"URL":"https:\/\/edm.bioscientifica.com\/downloadpdf\/journals\/edm\/2015\/1\/EDM15-0062.xml","content-type":"unspecified","content-version":"vor","intended-application":"similarity-checking"}],"deposited":{"date-parts":[[2026,7,20]],"date-time":"2026-07-20T13:46:08Z","timestamp":1784555168000},"score":1,"resource":{"primary":{"URL":"http:\/\/journals.bioscientifica.com\/edm\/article\/3372"}},"subtitle":[],"short-title":[],"issued":{"date-parts":[[2015,10,1]]},"references-count":24,"URL":"https:\/\/doi.org\/10.1530\/edm-15-0062","relation":{},"ISSN":["2052-0573"],"issn-type":[{"value":"2052-0573","type":"electronic"}],"subject":[],"published":{"date-parts":[[2015,10,1]]}}}