{"status":"ok","message-type":"work","message-version":"1.0.0","message":{"indexed":{"date-parts":[[2026,5,6]],"date-time":"2026-05-06T05:20:47Z","timestamp":1778044847356,"version":"3.51.4"},"reference-count":13,"publisher":"Springer Science and Business Media LLC","issue":"1","license":[{"start":{"date-parts":[[2023,2,16]],"date-time":"2023-02-16T00:00:00Z","timestamp":1676505600000},"content-version":"tdm","delay-in-days":0,"URL":"https:\/\/creativecommons.org\/licenses\/by\/4.0"},{"start":{"date-parts":[[2023,2,16]],"date-time":"2023-02-16T00:00:00Z","timestamp":1676505600000},"content-version":"vor","delay-in-days":0,"URL":"https:\/\/creativecommons.org\/licenses\/by\/4.0"}],"content-domain":{"domain":["link.springer.com"],"crossmark-restriction":false},"short-container-title":["BMC Endocr Disord"],"abstract":"<jats:title>Abstract<\/jats:title><jats:sec>\n                <jats:title>Background<\/jats:title>\n                <jats:p>Ectopic ACTH pituitary adenomas (EAPA), located outside the sella turcica and deriving from cellular remnants of Rathke\u2019s pouch are a very rare cause of Cushing\u2019s syndrome (CS). The diagnosis is often difficult and delayed, even after comprehensive work-up. To our knowledge, we report for the first time an ectopic corticotroph tumor of the posterior wall of the sphenoid sinus, leading to false positive results of bilateral inferior petrosal sinus sampling (BIPPS) and which was finally localized by a co-registered<jats:sup>11<\/jats:sup> C Methionine PET\/MR imaging.<\/jats:p>\n              <\/jats:sec><jats:sec>\n                <jats:title>Case presentation<\/jats:title>\n                <jats:p>A 48-year-old woman was referred for a high clinical suspicion of ACTH-dependent CS. Biological testing comprising low dose dexamethasone suppression and CRH stimulation tests were indicative of pituitary Cushing\u2019s disease, but comprehensive pituitary MRI did not reveal any pituitary adenoma. BIPSS confirmed however a central origin of ACTH secretion (central-to-peripheral ACTH ratio\u2009&gt;\u2009100) and revealed a significant right-to-left gradient (6.2), leading to a first right-sided exploratory hypophysectomy, that did not cure the patient. BIPSS images were reviewed and revealed preferential drainage of the left pituitary to the right petrosal sinus, leading us to a left sided exploratory hypophysectomy, which was again unsuccessful. A<jats:sup>11<\/jats:sup> C Methionine PET\/MRI was performed and revealed a hypermetabolic lesion adjacent to the posterior wall of the sphenoidal sinus. After surgical resection, this polypoid mass was identified as an ectopic ATCH-secreting pituitary adenoma expressing ACTH and T-Pit and complete remission of hypercortisolism was observed.<\/jats:p>\n              <\/jats:sec><jats:sec>\n                <jats:title>Conclusions<\/jats:title>\n                <jats:p>In conclusion, we report a case of ACTH-dependent Cushing\u2019s syndrome, caused by an ectopic corticotroph adenoma located in the sphenoidal sinus, which perfectly mimicked the biological features of a classical pituitary ACTH adenoma on a comprehensive hormonal evaluation including BIPPS, and the features of a benign naso-sinusal polyp at MRI. We report for the first time a key role of<jats:sup>11<\/jats:sup> C Methionine PET co-registered to high resolution MRI for localizing ectopic adenomas, efficiently guiding surgical removal and leading to complete remission of hypercortisolism.<\/jats:p>\n              <\/jats:sec>","DOI":"10.1186\/s12902-023-01298-2","type":"journal-article","created":{"date-parts":[[2023,2,16]],"date-time":"2023-02-16T09:02:59Z","timestamp":1676538179000},"update-policy":"https:\/\/doi.org\/10.1007\/springer_crossmark_policy","source":"Crossref","is-referenced-by-count":7,"title":["Ectopic sphenoidal ACTH-secreting adenoma revealed by 11C Methionine PET scan: case report"],"prefix":"10.1186","volume":"23","author":[{"given":"F.","family":"Lurquin","sequence":"first","affiliation":[],"role":[{"role":"author","vocabulary":"crossref"}]},{"ORCID":"https:\/\/orcid.org\/0000-0003-2665-9455","authenticated-orcid":false,"given":"S. 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Ectopic pituitary adenomas: clinical features, diagnostic challenges and management. Pituitary. 2020;23(6):648\u201364. https:\/\/doi.org\/10.1007\/s11102-020-01071-x.","journal-title":"Pituitary."},{"issue":"2","key":"1298_CR2","doi-asserted-by":"publisher","first-page":"E5","DOI":"10.3171\/2014.11.FOCUS14766","volume":"38","author":"F Pecori Giraldi","year":"2015","unstructured":"Pecori Giraldi F, Cavallo LM, Tortora F, et al. The role of inferior petrosal sinus sampling in ACTH-dependent Cushing\u2019s syndrome: review and joint opinion statement by members of the Italian Society for Endocrinology, Italian Society for Neurosurgery, and Italian Society for Neuroradiology. Neurosurg Focus. 2015;38(2):E5. https:\/\/doi.org\/10.3171\/2014.11.FOCUS14766.","journal-title":"Neurosurg Focus"},{"key":"1298_CR3","doi-asserted-by":"publisher","unstructured":"Mitsuhashi Y, Nishio A, Kawahara S, et al. 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Cushing\u2019s disease: pathobiology, diagnosis, and management. J Neurosurg. 2017;126(2):404\u201317. https:\/\/doi.org\/10.3171\/2016.1.JNS152119.","journal-title":"J Neurosurg"},{"issue":"6","key":"1298_CR6","doi-asserted-by":"publisher","first-page":"2285","DOI":"10.1210\/jc.2012-3943","volume":"98","author":"JJ Wind","year":"2013","unstructured":"Wind JJ, Lonser RR, Nieman LK, DeVroom HL, Chang R, Oldfield EH. The lateralization accuracy of inferior petrosal sinus sampling in 501 patients with cushing\u2019s disease. J Clin Endocrinol Metab. 2013;98(6):2285\u201393. https:\/\/doi.org\/10.1210\/jc.2012-3943.","journal-title":"J Clin Endocrinol Metab"},{"issue":"4","key":"1298_CR7","doi-asserted-by":"publisher","first-page":"M107","DOI":"10.1530\/EJE-15-0616","volume":"173","author":"O Koulouri","year":"2015","unstructured":"Koulouri O, Steuwe A, Gillett D, et al. A role for 11C-methionine PET imaging in ACTH-dependent Cushing\u2019s syndrome. 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