{"id":"https://openalex.org/W7223169587","doi":"https://doi.org/10.1210/jcemcr/luag323","title":"Severe insulin-requiring diabetes in a noradrenergic giant pheochromocytoma: a secretory-metabolic phenotype paradox","display_name":"Severe insulin-requiring diabetes in a noradrenergic giant pheochromocytoma: a secretory-metabolic phenotype paradox","publication_year":2026,"publication_date":"2026-10-16","ids":{"openalex":"https://openalex.org/W7223169587","doi":"https://doi.org/10.1210/jcemcr/luag323","pmid":"https://pubmed.ncbi.nlm.nih.gov/42923692","pmcid":"https://www.ncbi.nlm.nih.gov/pmc/articles/13687882"},"language":"en","primary_location":{"id":"doi:10.1210/jcemcr/luag323","doi":"https://doi.org/10.1210/jcemcr/luag323","is_oa":true,"landing_page_url":"https://doi.org/10.1210/jcemcr/luag323","pdf_url":"https://academic.oup.com/jcemcr/article-pdf/4/12/luag323/72330022/luag323.pdf","source":{"id":"https://openalex.org/S4387285112","display_name":"JCEM Case Reports","issn_l":"2755-1520","issn":["2755-1520"],"is_oa":true,"is_in_doaj":true,"is_core":true,"listed_in":["cwts-core","doaj","norway-1","vabb-shw"],"host_organization":null,"host_organization_name":null,"host_organization_lineage":[],"host_organization_lineage_names":[],"type":"journal"},"license":"cc-by","license_id":"https://openalex.org/licenses/cc-by","version":"publishedVersion","is_accepted":true,"is_published":true,"raw_source_name":"JCEM Case Reports","raw_type":"journal-article"},"volume":null,"type":"article","is_traditional":true,"indexed_in":["crossref","doaj","pubmed"],"open_access":{"is_oa":true,"oa_status":"gold","oa_url":"https://academic.oup.com/jcemcr/article-pdf/4/12/luag323/72330022/luag323.pdf","any_repository_has_fulltext":true},"authorships":[{"author_position":"first","author":{"id":"https://openalex.org/A5108862148","display_name":"Luiz F. Viola","orcid":"https://orcid.org/0000-0003-1167-7933","observed_orcids":["https://orcid.org/0000-0003-1167-7933"]},"institutions":[{"id":"https://openalex.org/I4210147289","display_name":"H\u00f4pital NOVO","ror":"https://ror.org/05jm9mh74","country_code":"FR","type":"healthcare","lineage":["https://openalex.org/I4210147289"]}],"countries":["FR"],"is_corresponding":true,"raw_author_name":"Luiz F Viola","raw_affiliation_strings":["Department of Endocrinology, Diabetes and Nutrition, NOVO Hospital , Pontoise 95300 ,"],"raw_orcid":"https://orcid.org/0000-0003-1167-7933","affiliations":[{"raw_affiliation_string":"Department of Endocrinology, Diabetes and Nutrition, NOVO Hospital , Pontoise 95300 ,","institution_ids":["https://openalex.org/I4210147289"]}]},{"author_position":"middle","author":{"id":"https://openalex.org/A5159240099","display_name":"Asma Guedioura","orcid":null,"observed_orcids":[]},"institutions":[{"id":"https://openalex.org/I4210147289","display_name":"H\u00f4pital NOVO","ror":"https://ror.org/05jm9mh74","country_code":"FR","type":"healthcare","lineage":["https://openalex.org/I4210147289"]}],"countries":["FR"],"is_corresponding":false,"raw_author_name":"Asma Guedioura","raw_affiliation_strings":["Department of Endocrinology, Diabetes and Nutrition, NOVO Hospital , Pontoise 95300 ,"],"raw_orcid":null,"affiliations":[{"raw_affiliation_string":"Department of Endocrinology, Diabetes and Nutrition, NOVO Hospital , Pontoise 95300 ,","institution_ids":["https://openalex.org/I4210147289"]}]},{"author_position":"middle","author":{"id":"https://openalex.org/A5006316711","display_name":"Josiane Neves Coelho Marques","orcid":"https://orcid.org/0009-0005-7330-7171","observed_orcids":["https://orcid.org/0009-0005-7330-7171"]},"institutions":[{"id":"https://openalex.org/I4396570648","display_name":"Universidade Federal de Rondon\u00f3polis","ror":"https://ror.org/044wn2t24","country_code":"BR","type":"education","lineage":["https://openalex.org/I4396570648"]}],"countries":["BR"],"is_corresponding":false,"raw_author_name":"Josiane Neves Coelho","raw_affiliation_strings":["Faculty of Health Sciences, Federal University of Rondon\u00f3polis , Rondon\u00f3polis 78736-900 ,"],"raw_orcid":"https://orcid.org/0009-0005-7330-7171","affiliations":[{"raw_affiliation_string":"Faculty of Health Sciences, Federal University of Rondon\u00f3polis , Rondon\u00f3polis 78736-900 ,","institution_ids":["https://openalex.org/I4396570648"]}]},{"author_position":"middle","author":{"id":"https://openalex.org/A5012852610","display_name":"Adriana Santi","orcid":"https://orcid.org/0000-0002-3681-5899","observed_orcids":["https://orcid.org/0000-0002-3681-5899"]},"institutions":[{"id":"https://openalex.org/I4396570648","display_name":"Universidade Federal de Rondon\u00f3polis","ror":"https://ror.org/044wn2t24","country_code":"BR","type":"education","lineage":["https://openalex.org/I4396570648"]}],"countries":["BR"],"is_corresponding":false,"raw_author_name":"Adriana Santi","raw_affiliation_strings":["Faculty of Health Sciences, Federal University of Rondon\u00f3polis , Rondon\u00f3polis 78736-900 ,"],"raw_orcid":"https://orcid.org/0000-0002-3681-5899","affiliations":[{"raw_affiliation_string":"Faculty of Health Sciences, Federal University of Rondon\u00f3polis , Rondon\u00f3polis 78736-900 ,","institution_ids":["https://openalex.org/I4396570648"]}]},{"author_position":"last","author":{"id":"https://openalex.org/A5036928342","display_name":"Catherine Campinos","orcid":null,"observed_orcids":[]},"institutions":[{"id":"https://openalex.org/I4210147289","display_name":"H\u00f4pital NOVO","ror":"https://ror.org/05jm9mh74","country_code":"FR","type":"healthcare","lineage":["https://openalex.org/I4210147289"]}],"countries":["FR"],"is_corresponding":false,"raw_author_name":"Catherine Campinos","raw_affiliation_strings":["Department of Endocrinology, Diabetes and Nutrition, NOVO Hospital , Pontoise 95300 ,"],"raw_orcid":null,"affiliations":[{"raw_affiliation_string":"Department of Endocrinology, Diabetes and Nutrition, NOVO Hospital , Pontoise 95300 ,","institution_ids":["https://openalex.org/I4210147289"]}]}],"institutions":[],"countries_distinct_count":2,"institutions_distinct_count":2,"corresponding_author_ids":["https://openalex.org/A5108862148"],"corresponding_institution_ids":["https://openalex.org/I4210147289"],"apc_list":{"value":930,"currency":"USD","value_usd":930},"apc_paid":{"value":930,"currency":"USD","value_usd":930},"fwci":0.0,"has_fulltext":true,"cited_by_count":0,"citation_normalized_percentile":{"value":0.0,"is_in_top_1_percent":false,"is_in_top_10_percent":false},"cited_by_percentile_year":null,"biblio":{"volume":"4","issue":"12","first_page":"luag323","last_page":"luag323"},"is_retracted":false,"is_paratext":false,"is_xpac":false,"primary_topic":{"id":"https://openalex.org/T11054","display_name":"Adrenal and Paraganglionic Tumors","score":0.9980000257492065,"subfield":{"id":"https://openalex.org/subfields/2746","display_name":"Surgery"},"field":{"id":"https://openalex.org/fields/27","display_name":"Medicine"},"domain":{"id":"https://openalex.org/domains/4","display_name":"Health Sciences"}},"topics":[{"id":"https://openalex.org/T11054","display_name":"Adrenal and Paraganglionic Tumors","score":0.9980000257492065,"subfield":{"id":"https://openalex.org/subfields/2746","display_name":"Surgery"},"field":{"id":"https://openalex.org/fields/27","display_name":"Medicine"},"domain":{"id":"https://openalex.org/domains/4","display_name":"Health Sciences"}},{"id":"https://openalex.org/T10754","display_name":"Neuroendocrine Tumor Research Advances","score":0.00039999998989515007,"subfield":{"id":"https://openalex.org/subfields/2730","display_name":"Oncology"},"field":{"id":"https://openalex.org/fields/27","display_name":"Medicine"},"domain":{"id":"https://openalex.org/domains/4","display_name":"Health Sciences"}},{"id":"https://openalex.org/T10839","display_name":"Pancreatic function and diabetes","score":0.0003000000142492354,"subfield":{"id":"https://openalex.org/subfields/2712","display_name":"Endocrinology, Diabetes and Metabolism"},"field":{"id":"https://openalex.org/fields/27","display_name":"Medicine"},"domain":{"id":"https://openalex.org/domains/4","display_name":"Health Sciences"}}],"keywords":[{"id":"https://openalex.org/keywords/pheochromocytoma","display_name":"pheochromocytoma","score":0.9580000042915344},{"id":"https://openalex.org/keywords/insulin-treated-diabetes","display_name":"insulin-treated diabetes","score":0.9670000076293945},{"id":"https://openalex.org/keywords/hypertension","display_name":"hypertension","score":0.5199999809265137},{"id":"https://openalex.org/keywords/postoperative-hypoglycemia","display_name":"postoperative hypoglycemia","score":0.8420000076293945},{"id":"https://openalex.org/keywords/adrenalectomy","display_name":"adrenalectomy","score":0.703000009059906},{"id":"https://openalex.org/keywords/normetanephrine","display_name":"normetanephrine","score":0.8059999942779541}],"concepts":[{"id":"https://openalex.org/C2779505177","wikidata":"https://www.wikidata.org/wiki/Q517109","display_name":"Normetanephrine","level":3,"score":0.8396000266075134},{"id":"https://openalex.org/C71924100","wikidata":"https://www.wikidata.org/wiki/Q11190","display_name":"Medicine","level":0,"score":0.8306999802589417},{"id":"https://openalex.org/C126322002","wikidata":"https://www.wikidata.org/wiki/Q11180","display_name":"Internal medicine","level":1,"score":0.7796000242233276},{"id":"https://openalex.org/C2779512018","wikidata":"https://www.wikidata.org/wiki/Q536269","display_name":"Pheochromocytoma","level":2,"score":0.7615000009536743},{"id":"https://openalex.org/C134018914","wikidata":"https://www.wikidata.org/wiki/Q162606","display_name":"Endocrinology","level":1,"score":0.7358999848365784},{"id":"https://openalex.org/C555293320","wikidata":"https://www.wikidata.org/wiki/Q12206","display_name":"Diabetes mellitus","level":2,"score":0.635699987411499},{"id":"https://openalex.org/C2775894120","wikidata":"https://www.wikidata.org/wiki/Q271663","display_name":"Catecholamine","level":2,"score":0.5993000268936157},{"id":"https://openalex.org/C2777538456","wikidata":"https://www.wikidata.org/wiki/Q311213","display_name":"Glycated hemoglobin","level":4,"score":0.5259000062942505},{"id":"https://openalex.org/C77411442","wikidata":"https://www.wikidata.org/wiki/Q181100","display_name":"Urinary system","level":2,"score":0.48179998993873596},{"id":"https://openalex.org/C2780668416","wikidata":"https://www.wikidata.org/wiki/Q202758","display_name":"Hypoglycemia","level":3,"score":0.46700000762939453},{"id":"https://openalex.org/C2780087391","wikidata":"https://www.wikidata.org/wiki/Q366774","display_name":"Adrenalectomy","level":2,"score":0.391400009393692},{"id":"https://openalex.org/C2779306644","wikidata":"https://www.wikidata.org/wiki/Q2002370","display_name":"Insulin","level":2,"score":0.3849000036716461},{"id":"https://openalex.org/C2776783082","wikidata":"https://www.wikidata.org/wiki/Q412766","display_name":"Metanephrine","level":3,"score":0.3668000102043152},{"id":"https://openalex.org/C158453852","wikidata":"https://www.wikidata.org/wiki/Q4684738","display_name":"Adrenergic","level":3,"score":0.3612000048160553},{"id":"https://openalex.org/C2777391703","wikidata":"https://www.wikidata.org/wiki/Q1053470","display_name":"Insulin resistance","level":3,"score":0.3578000068664551},{"id":"https://openalex.org/C2775859304","wikidata":"https://www.wikidata.org/wiki/Q132621","display_name":"Epinephrine","level":2,"score":0.33480000495910645},{"id":"https://openalex.org/C2777433750","wikidata":"https://www.wikidata.org/wiki/Q189580","display_name":"Acromegaly","level":4,"score":0.32760000228881836},{"id":"https://openalex.org/C2778514916","wikidata":"https://www.wikidata.org/wiki/Q186242","display_name":"Norepinephrine","level":3,"score":0.31189998984336853},{"id":"https://openalex.org/C2777180221","wikidata":"https://www.wikidata.org/wiki/Q3025883","display_name":"Type 2 diabetes","level":3,"score":0.28700000047683716},{"id":"https://openalex.org/C2780233176","wikidata":"https://www.wikidata.org/wiki/Q1939330","display_name":"Primary aldosteronism","level":3,"score":0.273499995470047},{"id":"https://openalex.org/C2777581072","wikidata":"https://www.wikidata.org/wiki/Q987319","display_name":"Secondary hypertension","level":3,"score":0.26249998807907104},{"id":"https://openalex.org/C136502266","wikidata":"https://www.wikidata.org/wiki/Q7226667","display_name":"Polyol pathway","level":4,"score":0.2605000138282776}],"mesh":[],"locations_count":3,"locations":[{"id":"doi:10.1210/jcemcr/luag323","doi":"https://doi.org/10.1210/jcemcr/luag323","is_oa":true,"landing_page_url":"https://doi.org/10.1210/jcemcr/luag323","pdf_url":"https://academic.oup.com/jcemcr/article-pdf/4/12/luag323/72330022/luag323.pdf","source":{"id":"https://openalex.org/S4387285112","display_name":"JCEM Case Reports","issn_l":"2755-1520","issn":["2755-1520"],"is_oa":true,"is_in_doaj":true,"is_core":true,"listed_in":["cwts-core","doaj","norway-1","vabb-shw"],"host_organization":null,"host_organization_name":null,"host_organization_lineage":[],"host_organization_lineage_names":[],"type":"journal"},"license":"cc-by","license_id":"https://openalex.org/licenses/cc-by","version":"publishedVersion","is_accepted":true,"is_published":true,"raw_source_name":"JCEM Case Reports","raw_type":"journal-article"},{"id":"pmh:oai:pubmedcentral.nih.gov:13687882","doi":null,"is_oa":true,"landing_page_url":"https://pmc.ncbi.nlm.nih.gov/articles/PMC13687882/","pdf_url":"https://pmc.ncbi.nlm.nih.gov/articles/PMC13687882/pdf/luag323.pdf","source":{"id":"https://openalex.org/S2764455111","display_name":"PubMed Central","issn_l":null,"issn":null,"is_oa":true,"is_in_doaj":false,"is_core":false,"listed_in":[],"host_organization":"https://openalex.org/I1299303238","host_organization_name":"National Institutes of Health","host_organization_lineage":["https://openalex.org/I1299303238"],"host_organization_lineage_names":[],"type":"repository"},"license":"cc-by","license_id":"https://openalex.org/licenses/cc-by","version":"publishedVersion","is_accepted":true,"is_published":true,"raw_source_name":"JCEM Case Rep","raw_type":"Text"},{"id":"pmid:42923692","doi":null,"is_oa":false,"landing_page_url":"https://pubmed.ncbi.nlm.nih.gov/42923692","pdf_url":null,"source":{"id":"https://openalex.org/S4306525036","display_name":"PubMed","issn_l":null,"issn":null,"is_oa":false,"is_in_doaj":false,"is_core":false,"listed_in":[],"host_organization":"https://openalex.org/I1299303238","host_organization_name":"National Institutes of Health","host_organization_lineage":["https://openalex.org/I1299303238"],"host_organization_lineage_names":[],"type":"repository"},"license":null,"license_id":null,"version":"publishedVersion","is_accepted":true,"is_published":true,"raw_source_name":"JCEM case reports","raw_type":"Case Reports"}],"best_oa_location":{"id":"doi:10.1210/jcemcr/luag323","doi":"https://doi.org/10.1210/jcemcr/luag323","is_oa":true,"landing_page_url":"https://doi.org/10.1210/jcemcr/luag323","pdf_url":"https://academic.oup.com/jcemcr/article-pdf/4/12/luag323/72330022/luag323.pdf","source":{"id":"https://openalex.org/S4387285112","display_name":"JCEM Case Reports","issn_l":"2755-1520","issn":["2755-1520"],"is_oa":true,"is_in_doaj":true,"is_core":true,"listed_in":["cwts-core","doaj","norway-1","vabb-shw"],"host_organization":null,"host_organization_name":null,"host_organization_lineage":[],"host_organization_lineage_names":[],"type":"journal"},"license":"cc-by","license_id":"https://openalex.org/licenses/cc-by","version":"publishedVersion","is_accepted":true,"is_published":true,"raw_source_name":"JCEM Case Reports","raw_type":"journal-article"},"sustainable_development_goals":[{"display_name":"Good health and well-being","id":"https://metadata.un.org/sdg/3","score":0.9940000176429749}],"study_designs":[{"display_name":"Case Report","id":"https://openalex.org/study-designs/case-report"}],"awards":[],"funders":[],"has_content":{"pdf":true,"grobid_xml":true},"content_urls":{"pdf":"https://content.openalex.org/works/W7223169587.pdf","grobid_xml":"https://content.openalex.org/works/W7223169587.grobid-xml"},"referenced_works_count":13,"referenced_works":["https://openalex.org/W1985934603","https://openalex.org/W2078257937","https://openalex.org/W2115406537","https://openalex.org/W2141460159","https://openalex.org/W2585589460","https://openalex.org/W2731564930","https://openalex.org/W3013803906","https://openalex.org/W3025592089","https://openalex.org/W3108748990","https://openalex.org/W3166085797","https://openalex.org/W4220716887","https://openalex.org/W7122610098","https://openalex.org/W7122684242"],"related_works":["https://openalex.org/W4403151088","https://openalex.org/W4413276947","https://openalex.org/W2141460159","https://openalex.org/W4409965727","https://openalex.org/W2012311864","https://openalex.org/W2743548441","https://openalex.org/W4387405374","https://openalex.org/W2885883530","https://openalex.org/W1969814398","https://openalex.org/W7098063323"],"abstract_inverted_index":{"Pheochromocytoma":[0],"is":[1],"a":[2,40,58,72,139],"rare":[3],"catecholamine-secreting":[4],"neuroendocrine":[5],"tumor":[6],"associated":[7],"with":[8,25,39,75,169],"hypertension":[9,53],"and":[10,45,52,95,101,112,132,141,148,172,177],"dysglycemia":[11],"in":[12,23],"20%":[13],"to":[14,30,56,156,195],"40%":[15],"of":[16,33,49,127,158,166,185],"cases.":[17],"Insulin-requiring":[18],"diabetes":[19,51,168],"has":[20],"been":[21],"reported":[22],"tumors":[24],"an":[26,46,104],"adrenergic":[27,170],"profile,":[28,74],"owing":[29],"epinephrine-mediated":[31],"inhibition":[32],"\u03b2-cell":[34,149],"secretion.":[35],"A":[36,123],"37-year-old":[37],"man":[38],"normal":[41,96,130],"body":[42],"mass":[43,61],"index":[44],"18-year":[47],"history":[48],"insulin-requiring":[50],"was":[54,110,154],"found":[55],"have":[57],"right":[59],"adrenal":[60],"measuring":[62],"10.1":[63],"\u00d7":[64,66],"8.0":[65],"6.7":[67],"cm.":[68],"Biochemical":[69],"evaluation":[70],"showed":[71],"noradrenergic":[73],"urinary":[76,97],"normetanephrine":[77],"greater":[78],"than":[79,188],"5000":[80],"\u03bcg/24":[81,90],"hours":[82,91],"(SI:":[83,118],">26":[84],"400":[85],"nmol/day;":[86],"reference":[87],"range,":[88],"70-538":[89],"[SI:":[92],"370-2841":[93],"nmol/day])":[94],"metanephrine.":[98],"Right":[99],"adrenalectomy":[100],"histopathology":[102],"confirmed":[103],"intra-adrenal":[105],"pheochromocytoma.":[106],"After":[107],"surgery,":[108],"insulin":[109,125,146],"discontinued,":[111],"glycated":[113],"hemoglobin":[114],"remained":[115],"below":[116],"6.1%":[117],"41-43":[119],"mmol/mol)":[120],"without":[121],"therapy.":[122],"preoperative":[124],"requirement":[126],"0.83":[128],"units/kg/day,":[129],"C-peptide,":[131],"negative":[133],"glutamic":[134],"acid":[135],"decarboxylase":[136],"antibodies":[137],"supported":[138],"mixed":[140],"reversible":[142],"mechanism":[143],"involving":[144],"catecholamine-induced":[145],"resistance":[147],"suppression.":[150],"Severe":[151],"postoperative":[152],"hypoglycemia":[153],"attributed":[155],"reversal":[157],"catecholamine":[159,175,197],"excess.":[160],"This":[161],"case":[162],"challenges":[163],"the":[164],"association":[165],"severe":[167],"pheochromocytomas":[171],"suggests":[173],"that":[174],"burden":[176],"exposure":[178,194],"duration":[179],"may":[180],"be":[181],"more":[182],"important":[183],"determinants":[184],"metabolic":[186],"severity":[187],"secretory":[189],"profile":[190],"alone,":[191],"after":[192],"prolonged":[193],"high":[196],"concentrations.":[198]},"counts_by_year":[],"updated_date":"2026-11-08T12:12:28.789561","created_date":"2026-10-22T00:00:00"}
