{"status":"ok","message-type":"work","message-version":"1.0.0","message":{"indexed":{"date-parts":[[2026,4,16]],"date-time":"2026-04-16T21:41:10Z","timestamp":1776375670231,"version":"3.51.2"},"reference-count":45,"publisher":"The Endocrine Society","issue":"4","license":[{"start":{"date-parts":[[2026,4,16]],"date-time":"2026-04-16T00:00:00Z","timestamp":1776297600000},"content-version":"vor","delay-in-days":28,"URL":"https:\/\/creativecommons.org\/licenses\/by\/4.0\/"}],"content-domain":{"domain":[],"crossmark-restriction":false},"short-container-title":[],"published-print":{"date-parts":[[2026,3,19]]},"abstract":"<jats:title>Abstract<\/jats:title>\n                  <jats:p>Acromegaly caused by ectopic growth hormone\u2013releasing hormone (GHRH)-secreting neuroendocrine tumor (NET) is extremely rare, with cosecreting NETs even more seldom. We report a case of a female patient who presented with primary hyperparathyroidism (pHPT) and a GHRH- and insulin cosecreting pancreatic NET (pNET) and genetically confirmed multiple endocrine neoplasia type 1 (MEN1), within an undiagnosed family with various MEN1-related NETs. Due to the diagnosis of MEN1, screening for pituitary tumor was performed with biochemical evidence of acromegaly. Sellar magnetic resonance imaging revealed a sellar lesion, which was excised and compatible with somatotroph hyperplasia. Postoperatively, the patient developed severe hypoglycemia requiring hospitalization and the pNET was removed. Histopathology confirmed GHRH and insulin secreting pNET. Ectopic acromegaly in MEN1 is exceedingly rare. Patients with MEN1 often present with multiple pNETs, which may exhibit multihormonal secretion and frequently cosecrete GHRH and insulin in MEN1, while hypoglycemia may not be manifested possibly due to GH and insulin's counteractive effects on glucose metabolism.<\/jats:p>","DOI":"10.1210\/jcemcr\/luag002","type":"journal-article","created":{"date-parts":[[2026,1,7]],"date-time":"2026-01-07T12:43:44Z","timestamp":1767789824000},"source":"Crossref","is-referenced-by-count":0,"title":["GHRH and insulin hypersecretion from a pancreatic neuroendocrine tumor in multiple endocrine neoplasia type 1"],"prefix":"10.1210","volume":"4","author":[{"ORCID":"https:\/\/orcid.org\/0000-0002-6026-4329","authenticated-orcid":false,"given":"Elisa","family":"Lamback","sequence":"first","affiliation":[{"name":"Neuroendocrinology Research Center, Endocrinology Section, Medical School and Hospital Universit\u00e1rio Clementino Fraga Filho, Universidade Federal do Rio de Janeiro , Rio de Janeiro 21941-617 ,","place":["Brazil"]},{"name":"Neuropathology and Molecular Genetics Laboratory, Instituto Estadual do C\u00e9rebro Paulo Niemeyer , Rio de Janeiro 20231-092 ,","place":["Brazil"]},{"name":"Neuroendocrine Unit, Instituto Estadual do C\u00e9rebro Paulo Niemeyer , Rio de Janeiro 20231-092 ,","place":["Brazil"]}],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Daniel Alves","family":"Bulzico","sequence":"additional","affiliation":[{"name":"Endocrine Oncology Unit, Brazilian National Institute of Cancer , Rio de Janeiro 20230-130 ,","place":["Brazil"]}],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Delmar Muniz","family":"Louren\u00e7o","sequence":"additional","affiliation":[{"name":"Endocrine Genetics Unit, Laboratory of Cellular and Molecular Endocrinology (LIM-25), Division of Endocrinology and Metabolism, Hospital das Cl\u00ednicas, University of S\u00e3o Paulo School of Medicine , S\u00e3o Paulo 05403-010 ,","place":["Brazil"]},{"name":"Division of Endocrine Oncology, Institute of Cancer of the State of S\u00e3o Paulo, University of S\u00e3o Paulo School of Medicine , S\u00e3o Paulo 01246-000 ,","place":["Brazil"]}],"role":[{"role":"author","vocabulary":"crossref"}]},{"ORCID":"https:\/\/orcid.org\/0000-0001-5954-0317","authenticated-orcid":false,"given":"Alexandre","family":"Vasiljevic","sequence":"additional","affiliation":[{"name":"Centre de Pathologie Est, Groupement Hospitalier Est, Hospices Civils de Lyon , Lyon 69003 ,","place":["France"]}],"role":[{"role":"author","vocabulary":"crossref"}]},{"ORCID":"https:\/\/orcid.org\/0000-0002-9517-338X","authenticated-orcid":false,"given":"Gerald","family":"Raverot","sequence":"additional","affiliation":[{"name":"Cancer Research Center of Lyon, Inserm U1052, Claude Bernard Lyon 1 University , Lyon 69373 ,","place":["France"]},{"name":"Endocrinology Department, Reference Center for Rare Pituitary Diseases HYPO, \u201cGroupement Hospitalier Est\u201d Hospices Civils de Lyon , Bron 69500 ,","place":["France"]}],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"M\u00f4nica R","family":"Gadelha","sequence":"additional","affiliation":[{"name":"Neuroendocrinology Research Center, Endocrinology Section, Medical School and Hospital Universit\u00e1rio Clementino Fraga Filho, Universidade Federal do Rio de Janeiro , Rio de Janeiro 21941-617 ,","place":["Brazil"]},{"name":"Neuropathology and Molecular Genetics Laboratory, Instituto Estadual do C\u00e9rebro Paulo Niemeyer , Rio de Janeiro 20231-092 ,","place":["Brazil"]},{"name":"Neuroendocrine Unit, Instituto Estadual do C\u00e9rebro Paulo Niemeyer , Rio de Janeiro 20231-092 ,","place":["Brazil"]}],"role":[{"role":"author","vocabulary":"crossref"}]}],"member":"80","published-online":{"date-parts":[[2026,4,16]]},"reference":[{"issue":"6","key":"2026041616411539500_luag002-B1","doi-asserted-by":"crossref","first-page":"497","DOI":"10.1016\/j.ando.2012.09.004","article-title":"Acromegaly induced by ectopic secretion of GHRH: a review 30 years after GHRH discovery","volume":"73","author":"Borson-Chazot","year":"2012","journal-title":"Ann Endocrinol (Paris)"},{"issue":"3","key":"2026041616411539500_luag002-B2","doi-asserted-by":"crossref","first-page":"101877","DOI":"10.1016\/j.beem.2024.101877","article-title":"The clinical and biochemical spectrum of ectopic acromegaly","volume":"38","author":"Fainstein-Day","year":"2024","journal-title":"Best Pract Res Clin Endocrinol Metab"},{"issue":"7","key":"2026041616411539500_luag002-B3","doi-asserted-by":"crossref","first-page":"609","DOI":"10.1159\/000511791","article-title":"Multiple endocrine neoplasia type 1 and the pancreas: diagnosis and treatment of functioning and non-functioning pancreatic and duodenal neuroendocrine neoplasia within the MEN1 syndrome - 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