{"status":"ok","message-type":"work","message-version":"1.0.0","message":{"indexed":{"date-parts":[[2026,7,8]],"date-time":"2026-07-08T05:21:02Z","timestamp":1783488062133,"version":"3.55.0"},"reference-count":26,"publisher":"Frontiers Media SA","license":[{"start":{"date-parts":[[2024,2,8]],"date-time":"2024-02-08T00:00:00Z","timestamp":1707350400000},"content-version":"vor","delay-in-days":0,"URL":"https:\/\/creativecommons.org\/licenses\/by\/4.0\/"}],"content-domain":{"domain":["frontiersin.org"],"crossmark-restriction":true},"short-container-title":["Front. Endocrinol."],"abstract":"<jats:p>This report describes a rare case of a 20-year-old man with an ACTH- and prolactin-secreting invasive pituitary macroadenoma causing hyperprolactinemia and Cushing\u2019s disease. He was later found to have an AIP mutation. Treatment with cabergoline (1.5 mg weekly) normalized prolactin concentrations and induced a major shrinkage of the adenoma. Not only was urinary free cortisol normalized for more than 14 years, but also the treatment induced normal hypothalamo-pituitary-adrenal (HPA) axis function as illustrated by the reappearance of a normal cortisol\/ACTH circadian rhythm, cortisol suppression to dexamethasone, and disappearance of the excessive and aberrant responses to CRH and desmopressin, respectively. This case is the first description of complete restoration of the physiological characteristics of the HPA axis by a medication during the treatment of Cushing\u2019s disease. Although exceptional, it illustrates that drugs targeting the pituitary adenoma can bring true complete remission of Cushing\u2019s disease.<\/jats:p>","DOI":"10.3389\/fendo.2024.1337741","type":"journal-article","created":{"date-parts":[[2024,2,8]],"date-time":"2024-02-08T05:19:07Z","timestamp":1707369547000},"update-policy":"https:\/\/doi.org\/10.3389\/crossmark-policy","source":"Crossref","is-referenced-by-count":5,"title":["Case report: Complete restoration of the HPA axis function in Cushing\u2019s disease with drug treatment"],"prefix":"10.3389","volume":"15","author":[{"given":"Joanne Thanh-T\u00e2m","family":"Nguyen","sequence":"first","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Amandine","family":"Ferri\u00e8re","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Antoine","family":"Tabarin","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]}],"member":"1965","published-online":{"date-parts":[[2024,2,8]]},"reference":[{"key":"B1","doi-asserted-by":"publisher","DOI":"10.1210\/jc.2015-1818","article-title":"Treatment of cushing\u2019s syndrome: an endocrine society clinical practice guideline","volume":"100","author":"Nieman","year":"2015","journal-title":"J Clin Endocrinol Metab"},{"key":"B2","doi-asserted-by":"publisher","DOI":"10.1016\/S2213-8587(21)00235-7","article-title":"Consensus on diagnosis and management of Cushing\u2019s disease: a guideline update","volume":"9","author":"Fleseriu","year":"2021","journal-title":"Lancet Diabetes Endocrinol"},{"key":"B3","doi-asserted-by":"publisher","DOI":"10.1210\/clinem\/dgad251","article-title":"Evidence of persistent mild hypercortisolism in patients medically treated for Cushing\u2019s disease: the Haircush study","author":"Mohammedi","year":"2023","journal-title":"J Clin Endocrinol Metab"},{"key":"B4","doi-asserted-by":"publisher","DOI":"10.1016\/j.ando.2018.03.012","article-title":"Are Cushing\u2019s disease patients curable","volume":"79","author":"Bertagna","year":"2018","journal-title":"Ann Endocrinol (Paris)"},{"key":"B5","doi-asserted-by":"publisher","DOI":"10.1530\/EJE-19-0695","article-title":"Use of late-night salivary cortisol to monitor response to medical treatment in Cushing\u2019s disease","volume":"182","author":"Newell-Price","year":"2020","journal-title":"Eur J Endocrinol"},{"key":"B6","doi-asserted-by":"publisher","DOI":"10.1210\/er.2012-1013","article-title":"Familial isolated pituitary adenomas (FIPA) and the pituitary adenoma predisposition due to mutations in the aryl hydrocarbon receptor interacting protein (AIP) gene","volume":"34","author":"Beckers","year":"2013","journal-title":"Endocrine Rev"},{"key":"B7","doi-asserted-by":"publisher","DOI":"10.1210\/jc.2011-2291","article-title":"Germline AIP mutations in apparently sporadic pituitary adenomas: prevalence in a prospective single-center cohort of 443 patients","volume":"97","author":"Cazabat","year":"2012","journal-title":"J Clin Endocrinol Metab"},{"key":"B8","doi-asserted-by":"publisher","DOI":"10.1111\/j.1399-0004.2010.01406.x","article-title":"The role of germline AIP, MEN1, PRKAR1A, CDKN1B and CDKN2C mutations in causing pituitary adenomas in a large cohort of children, adolescents, and patients with genetic syndromes","volume":"78","author":"Stratakis","year":"2010","journal-title":"Clin Genet"},{"key":"B9","doi-asserted-by":"publisher","DOI":"10.3389\/fendo.2023.1098367","article-title":"AIP gene germline variants in adult Polish patients with apparently sporadic pituitary macroadenomas","volume":"14","author":"Trofimiuk-M\u00fcldner","year":"2023","journal-title":"Front Endocrinol"},{"key":"B10","doi-asserted-by":"publisher","first-page":"6","DOI":"10.1007\/s12022-022-09703-7","article-title":"Overview of the 2022 WHO classification of pituitary tumors","volume":"33","author":"Asa","year":"2022","journal-title":"Endocrine Pathology."},{"key":"B11","doi-asserted-by":"publisher","DOI":"10.1210\/jc.2009-2556","article-title":"Clinical characteristics and therapeutic responses in patients with germ-line AIP mutations and pituitary adenomas: an international collaborative study","volume":"95","author":"Daly","year":"2010","journal-title":"J Clin Endocrinol Metab"},{"key":"B12","doi-asserted-by":"publisher","DOI":"10.1056\/NEJMoa1105743","article-title":"A 12-month phase 3 study of pasireotide in Cushing\u2019s disease","volume":"366","author":"Colao","year":"2012","journal-title":"New Engl J Med"},{"key":"B13","doi-asserted-by":"publisher","first-page":"17","DOI":"10.1016\/S2213-8587(17)30326-1","article-title":"Efficacy and safety of once-monthly pasireotide in Cushing\u2019s disease: a 12 month clinical trial","volume":"6","author":"Lacroix","year":"2018","journal-title":"Lancet Diabetes Endocrinol"},{"key":"B14","doi-asserted-by":"publisher","DOI":"10.1530\/EJE-16-0662","article-title":"Cabergoline for Cushing\u2019s disease: a large retrospective multicenter study","volume":"176","author":"Ferriere","year":"2017","journal-title":"Eur J endocrinology."},{"key":"B15","doi-asserted-by":"publisher","DOI":"10.1210\/jc.2008-1533","article-title":"The medical treatment of Cushing\u2019s disease: effectiveness of chronic treatment with the dopamine agonist cabergoline in patients unsuccessfully treated by surgery","volume":"94","author":"Pivonello","year":"2009","journal-title":"J Clin Endocrinol Metab"},{"key":"B16","doi-asserted-by":"publisher","DOI":"10.1007\/BF03348376","article-title":"Cushing\u2019s disease and marked hyperprolactinemia in a patient with a pituitary macroadenoma: effectiveness of bromocriptine treatment","volume":"7","author":"Verde","year":"1984","journal-title":"J endocrinological Invest"},{"key":"B17","doi-asserted-by":"publisher","DOI":"10.1007\/BF03343983","article-title":"Macroprolactinoma associated with Cushing\u2019s disease, successfully treated with cabergoline","volume":"25","author":"T\u2019Sjoen","year":"2002","journal-title":"J endocrinological Invest"},{"key":"B18","doi-asserted-by":"publisher","DOI":"10.1136\/bcr-2017-219921","article-title":"An unusual case of Cushing\u2019s syndrome due to bihormonal ACTH-prolactin secreting pituitary macroadenoma with rapid response to cabergoline","volume":"2017","author":"Kunasegaran","year":"2017","journal-title":"BMJ Case Rep"},{"key":"B19","doi-asserted-by":"publisher","DOI":"10.2169\/internalmedicine.55.5301","article-title":"Concomitant cushing\u2019s disease and marked hyperprolactinemia: response to a dopamine receptor agonist","volume":"55","author":"Shiraishi","year":"2016","journal-title":"Internal Med (Tokyo Japan)."},{"key":"B20","doi-asserted-by":"publisher","DOI":"10.1210\/jcem-55-5-947","article-title":"Concurrent production of adrenocorticotropin and prolactin from two distinct cell lines in a single pituitary adenoma: a detailed immunohistochemical analysis","volume":"55","author":"Sherry","year":"1982","journal-title":"J Clin Endocrinol Metab"},{"key":"B21","doi-asserted-by":"publisher","first-page":"598","DOI":"10.1016\/S0344-0338(11)80153-6","article-title":"Cushing\u2019s disease and hyperprolactinemia due to a mixed ACTH- and prolactin-secreting pituitary macroadenoma","volume":"187","author":"Mahler","year":"1991","journal-title":"Pathology Res practice."},{"key":"B22","doi-asserted-by":"publisher","DOI":"10.1007\/BF03343688","article-title":"From macroprolactinoma to concomitant ACTH-PRL hypersecretion with Cushing\u2019s disease","volume":"23","author":"Barausse","year":"2000","journal-title":"J endocrinological Invest"},{"key":"B23","doi-asserted-by":"publisher","DOI":"10.1530\/EDM-17-0027","article-title":"Hypersecretion of ACTH and PRL from pituitary adenoma in MEN1, adequately managed by medical therapy","volume":"2017","author":"Uraki","year":"2017","journal-title":"Endocrinology Diabetes Metab Case Rep"},{"key":"B24","doi-asserted-by":"publisher","DOI":"10.1056\/NEJMc1204078","article-title":"Pasireotide in cushing\u2019s disease","volume":"366","author":"Lib\u00e9","year":"2012","journal-title":"New Engl J Med"},{"key":"B25","doi-asserted-by":"publisher","DOI":"10.1111\/cen.12534","article-title":"Accuracy of repeated measurements of late-night salivary cortisol to screen for early-stage recurrence of Cushing\u2019s disease following pituitary surgery","volume":"82","author":"Danet-Lamasou","year":"2015","journal-title":"Clin Endocrinology."},{"key":"B26","doi-asserted-by":"publisher","DOI":"10.1038\/s41574-023-00868-7","article-title":"An individualized approach to the management of Cushing disease","volume":"19","author":"Fleseriu","year":"2023","journal-title":"Nat Rev Endocrinology."}],"container-title":["Frontiers in Endocrinology"],"original-title":[],"link":[{"URL":"https:\/\/www.frontiersin.org\/articles\/10.3389\/fendo.2024.1337741\/full","content-type":"unspecified","content-version":"vor","intended-application":"similarity-checking"}],"deposited":{"date-parts":[[2024,2,8]],"date-time":"2024-02-08T05:19:12Z","timestamp":1707369552000},"score":1,"resource":{"primary":{"URL":"https:\/\/www.frontiersin.org\/articles\/10.3389\/fendo.2024.1337741\/full"}},"subtitle":[],"short-title":[],"issued":{"date-parts":[[2024,2,8]]},"references-count":26,"alternative-id":["10.3389\/fendo.2024.1337741"],"URL":"https:\/\/doi.org\/10.3389\/fendo.2024.1337741","relation":{},"ISSN":["1664-2392"],"issn-type":[{"value":"1664-2392","type":"electronic"}],"subject":[],"published":{"date-parts":[[2024,2,8]]},"article-number":"1337741"}}