{"status":"ok","message-type":"work","message-version":"1.0.0","message":{"indexed":{"date-parts":[[2026,10,10]],"date-time":"2026-10-10T03:20:29Z","timestamp":1791602429391,"version":"4.3.4"},"reference-count":36,"publisher":"The Endocrine Society","issue":"6","license":[{"start":{"date-parts":[[2022,12,6]],"date-time":"2022-12-06T00:00:00Z","timestamp":1670284800000},"content-version":"vor","delay-in-days":0,"URL":"https:\/\/creativecommons.org\/licenses\/by-nc-nd\/4.0\/"}],"content-domain":{"domain":[],"crossmark-restriction":false},"short-container-title":[],"published-print":{"date-parts":[[2023,5,17]]},"abstract":"<jats:title>Abstract<\/jats:title>\n                  <jats:sec>\n                    <jats:title>Context<\/jats:title>\n                    <jats:p>Prospective studies have demonstrated the efficacy of osilodrostat in Cushing disease. No study has evaluated osilodrostat in a series of patients with paraneoplastic Cushing syndrome\/ectopic adrenocorticotropin syndrome (PNCS\/EAS).<\/jats:p>\n                  <\/jats:sec>\n                  <jats:sec>\n                    <jats:title>Objective<\/jats:title>\n                    <jats:p>This work aimed to evaluate in France the real-world efficacy and safety of osilodrostat in patients with PNCS\/EAS.<\/jats:p>\n                  <\/jats:sec>\n                  <jats:sec>\n                    <jats:title>Methods<\/jats:title>\n                    <jats:p>A total of 33 patients with PNCS\/EAS with intense\/severe hypercortisolism were involved in this retrospective, multicenter, real-world study. Patients received osilodrostat between May 2019 and March 2022 at a median initial dose (range) of 4 mg\/day (1-60) and maximum dose, 20 mg\/day (4-100), first under patient then cohort temporary authorizations and after marketing authorization. Regimens used titration (n = 6), block and replace (n = 16), or titration followed by block and replace (n = 11).<\/jats:p>\n                  <\/jats:sec>\n                  <jats:sec>\n                    <jats:title>Results<\/jats:title>\n                    <jats:p>In 11 patients receiving osilodrostat as first-line monotherapy, median 24-hour urinary free cortisol (24h-UFC) decreased dramatically (from 26 \u00d7 upper limit of normal [ULN; 2.9-659] to 0.11 \u00d7 ULN [0.08-14.9]; P &amp;lt; .001). In 9 of them, 24h-UFC normalization was achieved in 2 weeks (median). Thirteen additional patients were previously treated with classic steroidogenesis inhibitors but 10 of these 13 were not controlled. In these patients, osilodrostat monotherapy, used as second line, induced a significantly decreased of 24h-UFC (from 2.6 \u00d7 ULN [1.1-144] to 0.22 \u00d7 ULN [0.12-0.66]; P &amp;lt; .01). Nine additional patients received osilodrostat in combination with another anticortisolic drug, decreasing 24h-UFC from 11.8 \u00d7 ULN (0.3-247) to 0.43 \u00d7 ULN (0.33-2.4) (P &amp;lt; .01). In parallel, major clinical symptoms\/comorbidities improved dramatically with improvement in blood pressure, hyperglycemia, and hypokalemia, allowing the discontinuation or dose reduction of patient treatments. Adrenal insufficiency (grade 3-4) was reported in 8 of 33 patients.<\/jats:p>\n                  <\/jats:sec>\n                  <jats:sec>\n                    <jats:title>Conclusion<\/jats:title>\n                    <jats:p>Osilodrostat is a rapidly efficient therapy for PNCS\/EAS with severe\/intense hypercortisolism. Osilodrostat was generally well tolerated; adrenal insufficiency was the main side effect.<\/jats:p>\n                  <\/jats:sec>","DOI":"10.1210\/clinem\/dgac691","type":"journal-article","created":{"date-parts":[[2022,12,6]],"date-time":"2022-12-06T00:19:29Z","timestamp":1670285969000},"page":"1475-1487","source":"Crossref","is-referenced-by-count":69,"title":["Efficacy and Safety of Osilodrostat in Paraneoplastic Cushing Syndrome: A Real-World Multicenter Study in France"],"prefix":"10.1210","volume":"108","author":[{"given":"Alexandre","family":"Dormoy","sequence":"first","affiliation":[{"name":"Paris-Saclay University; Assistance Publique-H\u00f4pitaux de Paris, Department of Endocrinology, Reference Centre for Rare Pituitary Diseases HYPO, Bic\u00eatre Hospital , Le Kremlin-Bic\u00eatre, F-94275 , France"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Magalie","family":"Haissaguerre","sequence":"additional","affiliation":[{"name":"Bordeaux University, Department of Endocrinology, Haut-L\u00e9v\u00eaque Hospital , F-33600, Pessac , France"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"G\u00e9raldine","family":"Vitellius","sequence":"additional","affiliation":[{"name":"Department of Endocrinology, Robert Debr\u00e9 University Hospital , F-51100, Reims , France"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Christine","family":"Do Cao","sequence":"additional","affiliation":[{"name":"Department of Endocrinology, Centre Hospitalier R\u00e9gional Universitaire de Lille , F-59037, Lille , France"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Aurore","family":"Geslot","sequence":"additional","affiliation":[{"name":"Department of Endocrinology and Metabolic Diseases, Larrey University Hospital , F-31059, Toulouse , France"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Delphine","family":"Drui","sequence":"additional","affiliation":[{"name":"Department of Endocrinology, Institut du Thorax, CHU de Nantes, and Nantes Universit\u00e9, H\u00f4pital Nord , F-44000 Nantes , France"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0000-0002-5027-7660","authenticated-orcid":false,"given":"H\u00e9l\u00e8ne","family":"Lasolle","sequence":"additional","affiliation":[{"name":"Endocrinology Department, Reference Centre for Rare Pituitary Diseases HYPO, \u201cGroupement Hospitalier Est\u201d Hospices Civils de Lyon , F-69500 Bron , France"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Oceana","family":"Vieira-Pinto","sequence":"additional","affiliation":[{"name":"Paris-Saclay University; Assistance Publique-H\u00f4pitaux de Paris, Department of Endocrinology, Reference Centre for Rare Pituitary Diseases HYPO, Bic\u00eatre Hospital , Le Kremlin-Bic\u00eatre, F-94275 , France"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Sylvie","family":"Salenave","sequence":"additional","affiliation":[{"name":"Paris-Saclay University; Assistance Publique-H\u00f4pitaux de Paris, Department of Endocrinology, Reference Centre for Rare Pituitary Diseases HYPO, Bic\u00eatre Hospital , Le Kremlin-Bic\u00eatre, F-94275 , France"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Maud","family":"Fran\u00e7ois","sequence":"additional","affiliation":[{"name":"Department of Endocrinology, Robert Debr\u00e9 University Hospital , F-51100, Reims , France"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Marie","family":"Puerto","sequence":"additional","affiliation":[{"name":"Bordeaux University, Department of Endocrinology, Haut-L\u00e9v\u00eaque Hospital , F-33600, Pessac , France"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"H\u00e9l\u00e8ne Du","family":"Boullay","sequence":"additional","affiliation":[{"name":"Department of Endocrinology, Savoie CHMS Hospital , F-73000 Chamb\u00e9ry , France"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Anne","family":"Mayer","sequence":"additional","affiliation":[{"name":"Department of Endocrinology, Savoie CHMS Hospital , F-73000 Chamb\u00e9ry , France"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Anne","family":"Rod","sequence":"additional","affiliation":[{"name":"Department of Endocrinology, CH de Niort , F-79000, Niort , France"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Claire","family":"Laurent","sequence":"additional","affiliation":[{"name":"Department of Endocrinology, CH de Niort , F-79000, Niort , 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France"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0000-0002-1808-8800","authenticated-orcid":false,"given":"Fr\u00e9d\u00e9ric","family":"Castinetti","sequence":"additional","affiliation":[{"name":"Department of Endocrinology, Assistance Publique-Hopitaux de Marseille, French Reference Center for Rare Pituitary Diseases, Endo-European Reference Network and EURACAN European Expert Center on Rare Pituitary Tumors, La Conception Hospital, Aix Marseille University , F-13385, Marseille , France"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0000-0001-8756-7218","authenticated-orcid":false,"given":"Olivier","family":"Chabre","sequence":"additional","affiliation":[{"name":"University Grenoble Alpes, UMR 1292 INSERM-CEA-UGA, Endocrinologie CHU Grenoble Alpes , F-38000 Grenoble , 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