{"status":"ok","message-type":"work","message-version":"1.0.0","message":{"indexed":{"date-parts":[[2026,7,20]],"date-time":"2026-07-20T14:17:55Z","timestamp":1784557075022,"version":"3.55.0"},"reference-count":18,"publisher":"Bioscientifica","issue":"1","license":[{"start":{"date-parts":[[2023,1,1]],"date-time":"2023-01-01T00:00:00Z","timestamp":1672531200000},"content-version":"unspecified","delay-in-days":0,"URL":"http:\/\/creativecommons.org\/licenses\/by-nc-nd\/4.0\/"}],"content-domain":{"domain":["edm.bioscientifica.com"],"crossmark-restriction":true},"short-container-title":[],"published-print":{"date-parts":[[2023,1,1]]},"abstract":"<jats:sec>\n                    <jats:title>Summary<\/jats:title>\n                    <jats:p>\n                      A 64-year-old man with progressive metastatic castrate-resistant prostate adenocarcinoma presented with recurrent fluid overload, severe hypokalaemia with metabolic alkalosis and loss of glycaemic control. Clinical features were facial plethora, skin bruising and proximal myopathy. Plasma adrenocorticotrophic hormone (ACTH), serum cortisol and 24-h urinary cortisol levels were elevated. Low-dose dexamethasone failed to suppress cortisol. Pituitary MRI was normal and\n                      <jats:sup>68<\/jats:sup>\n                      Gallium-DOTATATE PET\u2013CT scan showed only features of metastatic prostate cancer. He was diagnosed with ectopic ACTH syndrome secondary to treatment-related neuroendocrine prostate cancer differentiation. Medical management was limited by clinical deterioration, accessibility of medications and cancer progression. Ketoconazole and cabergoline were utilised, but cortisol remained uncontrolled. He succumbed 5 months following diagnosis. Treatment-related neuroendocrine differentiation of prostate adenocarcinoma is a rare cause of ectopic ACTH syndrome.\n                    <\/jats:p>\n                  <\/jats:sec>\n                  <jats:sec>\n                    <jats:title>Learning points<\/jats:title>\n                    <jats:list list-type=\"bullet\">\n                      <jats:list-item>\n                        <jats:p>Neuroendocrine differentiation following prostate adenocarcinoma treatment with androgen deprivation has been described.<\/jats:p>\n                      <\/jats:list-item>\n                      <jats:list-item>\n                        <jats:p>Ectopic adrenocorticotrophic hormone (ACTH) syndrome should be considered where patients with metastatic prostate cancer develop acute electrolyte disturbance or fluid overload.<\/jats:p>\n                      <\/jats:list-item>\n                      <jats:list-item>\n                        <jats:p>Ketoconazole interferes with adrenal and gonadal steroidogenesis and can be used in ectopic ACTH syndrome, but the impact may be insufficient. Inhibition of gonadal steroidogenesis is favourable in prostate cancer.<\/jats:p>\n                      <\/jats:list-item>\n                      <jats:list-item>\n                        <jats:p>More data are required to evaluate the use of cabergoline in ectopic ACTH syndrome.<\/jats:p>\n                      <\/jats:list-item>\n                      <jats:list-item>\n                        <jats:p>Ectopic ACTH syndrome requires prompt management and is challenging in the face of metastatic cancer.<\/jats:p>\n                      <\/jats:list-item>\n                    <\/jats:list>\n                  <\/jats:sec>","DOI":"10.1530\/edm-22-0347","type":"journal-article","created":{"date-parts":[[2023,1,10]],"date-time":"2023-01-10T04:58:37Z","timestamp":1673326717000},"update-policy":"https:\/\/doi.org\/10.1530\/crossmarkpolicy-2","source":"Crossref","is-referenced-by-count":3,"title":["Ectopic adrenocorticotrophic hormone syndrome secondary to treatment-related neuroendocrine differentiation of metastatic castrate-resistant prostate cancer"],"prefix":"10.1530","volume":"2023","author":[{"ORCID":"https:\/\/orcid.org\/0000-0003-1802-8813","authenticated-orcid":true,"given":"Chelsea","family":"Tan","sequence":"first","affiliation":[{"name":"Bendigo Health, Victoria, Australia"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Jessica","family":"Triay","sequence":"additional","affiliation":[{"name":"Bendigo Health, Victoria, Australia"}],"role":[{"vocabulary":"crossref","role":"author"}]}],"member":"416","reference":[{"key":"bib1","series-title":"Journal of Clinical Oncology","first-page":"e386","article-title":"Challenges in recognizing treatment-related neuroendocrine prostate cancer","volume":"30","author":"Beltran","year":"2012","unstructured":"Beltran HTagawa STPark KMacDonald TMilowsky MIMosquera JMRubin MANanus DM. Challenges in recognizing treatment-related neuroendocrine prostate cancer. Journal of Clinical Oncology201230e386\u2013e389. (https:\/\/doi.org\/10.1200\/JCO.2011.41.5166)"},{"issue":"Supplement1","key":"bib2","series-title":"Translational Cancer Research","first-page":"S104","article-title":"Neuroendocrine differentiation in prostate cancer: key epigenetic players","volume":"6","author":"Gupta","year":"2017","unstructured":"Gupta KGupta S. Neuroendocrine differentiation in prostate cancer: key epigenetic players. Translational Cancer Research20176(Supplement1) S104\u2013S108. (https:\/\/doi.org\/10.21037\/tcr.2017.01.20)"},{"key":"bib3","series-title":"Endocrine Connections","first-page":"R80","article-title":"Severe Cushing\u2019s syndrome due to small cell prostate carcinoma: a case and review of literature","volume":"6","author":"Elston","year":"2017","unstructured":"Elston MSCrawford VBSwarbick MDray MSHead MConaglen JV. Severe Cushing\u2019s syndrome due to small cell prostate carcinoma: a case and review of literature. Endocrine Connections20176R80\u2013R86. (https:\/\/doi.org\/10.1530\/EC-17-0081)"},{"key":"bib4","series-title":"Annals of Laboratory Medicine","first-page":"521","article-title":"Second-line tests in the diagnosis of adrenocorticoptropic hormone-dependent hypercortisolism","volume":"41","author":"Pinelli","year":"2021","unstructured":"Pinelli SBarbot MScaroni CCeccato F. Second-line tests in the diagnosis of adrenocorticoptropic hormone-dependent hypercortisolism. Annals of Laboratory Medicine202141521\u2013531. (https:\/\/doi.org\/10.3343\/alm.2021.41.6.521)"},{"key":"bib5","series-title":"Clinical Endocrinology","first-page":"288","article-title":"The clinical impact of [68Ga]-DOTATATE PET\/CT for the diagnosis and management of ectopic adrenocorticotropic hormone \u2013 secreting tumors","volume":"91","author":"Wannachalee","year":"2019","unstructured":"Wannachalee TTurcu AFBancos IHabra MAAvram AMChuang HHWaguespack SGAuchus RJ. The clinical impact of [68Ga]-DOTATATE PET\/CT for the diagnosis and management of ectopic adrenocorticotropic hormone \u2013 secreting tumors. Clinical Endocrinology201991288\u2013294. (https:\/\/doi.org\/10.1111\/cen.14008)"},{"key":"bib6","series-title":"European Journal of Nuclear Medicine and Molecular Imaging","first-page":"4386","article-title":"Head-to-head comparison of 68Ga-DOTATATE PET\/CT and 18F-FDG PET\/CT in localizing tumors with ectopic adrenocorticotropic hormone secretion: a prospective study","volume":"48","author":"Liu","year":"2021","unstructured":"Liu QZang JYang YLing QWu HWang PLu LZhu Z. Head-to-head comparison of 68Ga-DOTATATE PET\/CT and 18F-FDG PET\/CT in localizing tumors with ectopic adrenocorticotropic hormone secretion: a prospective study. European Journal of Nuclear Medicine and Molecular Imaging2021484386\u20134395. (https:\/\/doi.org\/10.1007\/s00259-021-05370-8)"},{"key":"bib7","series-title":"European Journal of Endocrinology","first-page":"R29","article-title":"Management of endocrine disease. Cushing\u2019s syndrome due to ectopic ACTH secretion: an expert operational opinion","volume":"182","author":"Young","year":"2020","unstructured":"Young JHaissaguerre MViera-Pinto OChabre OBaudin ETabarin A. Management of endocrine disease. Cushing\u2019s syndrome due to ectopic ACTH secretion: an expert operational opinion. European Journal of Endocrinology2020182R29\u2013R58. (https:\/\/doi.org\/10.1530\/EJE-19-0877)"},{"key":"bib8","series-title":"European Journal of Endocrinology","first-page":"R263","article-title":"Therapy of endocrine disease. Steroidogenesis enzyme inhibitors in Cushing\u2019s syndrome","volume":"172","author":"Daniel","year":"2015","unstructured":"Daniel ENewell-Price JDC. Therapy of endocrine disease. Steroidogenesis enzyme inhibitors in Cushing\u2019s syndrome. European Journal of Endocrinology2015172R263\u2013R280. (https:\/\/doi.org\/10.1530\/EJE-14-1014)"},{"key":"bib9","series-title":"Journal of Clinical Endocrinology and Metabolism","first-page":"2807","article-title":"Treatment of Cushing\u2019s syndrome: an Endocrine Society clinical practice guideline","volume":"100","author":"Nieman","year":"2015","unstructured":"Nieman LKBiller BMKFindling JWMurad MHNewell-Price JSavage MOTabarin A & Endocrine Society. Treatment of Cushing\u2019s syndrome: an Endocrine Society clinical practice guideline. Journal of Clinical Endocrinology and Metabolism20151002807\u20132831. (https:\/\/doi.org\/10.1210\/jc.2015-1818)"},{"key":"bib10","series-title":"European Journal of Endocrinology","first-page":"473","article-title":"Rapid control of severe neoplastic hypercortisolism with metyrapone and ketoconazole","volume":"172","author":"Corcuff","year":"2015","unstructured":"Corcuff JBYoung JMasquefa-Giraud PChanson PBaudin ETabarin A. Rapid control of severe neoplastic hypercortisolism with metyrapone and ketoconazole. European Journal of Endocrinology2015172473\u2013481. (https:\/\/doi.org\/10.1530\/EJE-14-0913)"},{"key":"bib11","series-title":"European Journal of Endocrinology","first-page":"K1","article-title":"Cabergoline in severe ectopic or occult Cushing\u2019s syndrome","volume":"181","author":"Ilie","year":"2019","unstructured":"Ilie MDRaverot VTronc FVasiljevic ABorson-Chazot FRaverot G. Cabergoline in severe ectopic or occult Cushing\u2019s syndrome. European Journal of Endocrinology2019181K1\u2013K9. (https:\/\/doi.org\/10.1530\/EJE-18-1014)"},{"key":"bib12","series-title":"Endocrine Journal","first-page":"841","article-title":"A multicenter, phase 2 study to evaluate the efficacy and safety of osilodrostat, a new 11\u03b2-hydroxylase inhibitor, in Japanese patients with endogenous Cushing\u2019s syndrome other than Cushing\u2019s disease","volume":"67","author":"Tanaka","year":"2020","unstructured":"Tanaka TSatoh FUjihara MMidorikawa SKaneko TTakeda TSuzuki ASato MShimatsu A. A multicenter, phase 2 study to evaluate the efficacy and safety of osilodrostat, a new 11\u03b2-hydroxylase inhibitor, in Japanese patients with endogenous Cushing\u2019s syndrome other than Cushing\u2019s disease. Endocrine Journal202067841\u2013852. (https:\/\/doi.org\/10.1507\/endocrj.EJ19-0617)"},{"key":"bib13","series-title":"European Journal of Endocrinology","first-page":"L3","article-title":"Rapid control of severe ectopic Cushing\u2019s syndrome by oral orsilodrostat monotherapy","volume":"184","author":"Bessi\u00e8ne","year":"2021","unstructured":"Bessi\u00e8ne LBonnet FTenenbaum FJozwiak MCorchia ABertherat JGroussin L. Rapid control of severe ectopic Cushing\u2019s syndrome by oral orsilodrostat monotherapy. European Journal of Endocrinology2021184L3\u2013L15. (https:\/\/doi.org\/10.1530\/EJE-21-0147)"},{"key":"bib14","series-title":"European Journal of Endocrinology","first-page":"L7","article-title":"Efficacy and tolerance of osilodrostat in patients with severe Cushing\u2019s syndrome due to non-pituitary cancers","volume":"183","author":"Haissaguerre","year":"2020","unstructured":"Haissaguerre MPuerto MNunes MLTabarin A. Efficacy and tolerance of osilodrostat in patients with severe Cushing\u2019s syndrome due to non-pituitary cancers. European Journal of Endocrinology2020183L7\u2013L9. (https:\/\/doi.org\/10.1530\/EJE-20-0557)"},{"key":"bib15","series-title":"European Journal of Nuclear Medicine and Molecular Imaging","first-page":"1964","article-title":"The efficacy of 177Lu-labelled peptide receptor radionuclide therapy in patients with neuroendocrine tumours: a meta-analysis","volume":"42","author":"Kim","year":"2015","unstructured":"Kim SJPak KKoo PJKwak JJChang S. The efficacy of 177Lu-labelled peptide receptor radionuclide therapy in patients with neuroendocrine tumours: a meta-analysis. European Journal of Nuclear Medicine and Molecular Imaging2015421964\u20131970. (https:\/\/doi.org\/10.1007\/s00259-015-3155-x)"},{"key":"bib16","series-title":"Journal of Clinical Oncology","first-page":"2818","article-title":"Neuroendocrine tumors and peptide receptor radionuclide therapy: when is the right time?","volume":"40","author":"Hope","year":"2022","unstructured":"Hope TAPavel MBergsland EK. Neuroendocrine tumors and peptide receptor radionuclide therapy: when is the right time?Journal of Clinical Oncology2022402818\u20132829. (https:\/\/doi.org\/10.1200\/JCO.22.00176)"},{"key":"bib17","series-title":"European Journal of Nuclear Medicine and Molecular Imaging","first-page":"1831","article-title":"Assessment of predictors of response and long-term survival of patients with neuroendocrine tumour treated with peptide receptor chemoradionuclide therapy (PRCRT)","volume":"41","author":"Kong","year":"2014","unstructured":"Kong GThompson MCollins MHerschtal AHofman MSJohnston VEu PMichael MHicks RJ. Assessment of predictors of response and long-term survival of patients with neuroendocrine tumour treated with peptide receptor chemoradionuclide therapy (PRCRT). European Journal of Nuclear Medicine and Molecular Imaging2014411831\u20131844. (https:\/\/doi.org\/10.1007\/s00259-014-2788-5)"},{"key":"bib18","series-title":"AJR. American Journal of Roentgenology","first-page":"275","article-title":"Radioligand therapy with 177Lu-PSMA for metastatic castration-resistant prostate cancer: a systematic review and meta-analysis","volume":"213","author":"Yadav","year":"2019","unstructured":"Yadav MPBallal SSahoo RKDwivedi SNBal C. Radioligand therapy with 177Lu-PSMA for metastatic castration-resistant prostate cancer: a systematic review and meta-analysis. AJR. American Journal of Roentgenology2019213275\u2013285. (https:\/\/doi.org\/10.2214\/AJR.18.20845)"}],"container-title":["Endocrinology, Diabetes &amp; Metabolism Case Reports"],"original-title":[],"link":[{"URL":"https:\/\/edm.bioscientifica.com\/view\/journals\/edm\/2023\/1\/EDM22-0347.xml","content-type":"text\/html","content-version":"vor","intended-application":"text-mining"},{"URL":"https:\/\/edm.bioscientifica.com\/view\/journals\/edm\/2023\/1\/EDM22-0347.xml","content-type":"text\/html","content-version":"vor","intended-application":"syndication"},{"URL":"https:\/\/edm.bioscientifica.com\/view\/journals\/edm\/2023\/1\/EDM22-0347.xml","content-type":"unspecified","content-version":"vor","intended-application":"similarity-checking"}],"deposited":{"date-parts":[[2026,7,20]],"date-time":"2026-07-20T13:48:03Z","timestamp":1784555283000},"score":1,"resource":{"primary":{"URL":"http:\/\/journals.bioscientifica.com\/edm\/article\/3074"}},"subtitle":[],"short-title":[],"issued":{"date-parts":[[2023,1,1]]},"references-count":18,"journal-issue":{"issue":"1"},"URL":"https:\/\/doi.org\/10.1530\/edm-22-0347","relation":{},"ISSN":["2052-0573"],"issn-type":[{"value":"2052-0573","type":"electronic"}],"subject":[],"published":{"date-parts":[[2023,1,1]]}}}