{"status":"ok","message-type":"work","message-version":"1.0.0","message":{"indexed":{"date-parts":[[2026,11,7]],"date-time":"2026-11-07T05:11:22Z","timestamp":1794028282645,"version":"4.8.0"},"reference-count":22,"publisher":"Oxford University Press (OUP)","issue":"3","license":[{"start":{"date-parts":[[2026,9,30]],"date-time":"2026-09-30T00:00:00Z","timestamp":1790726400000},"content-version":"vor","delay-in-days":33,"URL":"https:\/\/creativecommons.org\/licenses\/by\/4.0\/"}],"funder":[{"DOI":"10.13039\/100017521","name":"ESTEVE","doi-asserted-by":"publisher","id":[{"id":"10.13039\/100017521","id-type":"DOI","asserted-by":"publisher"}]},{"name":"HRA Pharma Rare Diseases"}],"content-domain":{"domain":[],"crossmark-restriction":false},"short-container-title":[],"published-print":{"date-parts":[[2026,8,28]]},"abstract":"<jats:title>Abstract<\/jats:title>\n                  <jats:sec>\n                    <jats:title>Objective<\/jats:title>\n                    <jats:p>This analysis describes the evolution of health-related quality of life (HRQoL) over 36 weeks of metyrapone treatment in the PROMPT study, identifying patterns of early, delayed improvement, or persistence using item-level resolution.<\/jats:p>\n                  <\/jats:sec>\n                  <jats:sec>\n                    <jats:title>Design and Methods<\/jats:title>\n                    <jats:p>Longitudinal analysis of patient-reported outcomes from PROMPT (NCT02297945), a prospective, open-label, multicountry study of metyrapone in 49 evaluable adults with confirmed endogenous CS. HRQoL was assessed with the CushingQoL (0\u2013100; higher = better QoL) and T\u00fcbingen CD-25 (0\u2013100; higher = worse QoL) at weeks 4, 12, 24, and 36. Item-level analyses characterized the pattern and timing of improvement. Within-patient changes were assessed using the Wilcoxon signed-rank test.<\/jats:p>\n                  <\/jats:sec>\n                  <jats:sec>\n                    <jats:title>Results<\/jats:title>\n                    <jats:p>Both questionnaires demonstrated significant overall improvement by week 36: CushingQoL increased by 10.4 points (P &amp;lt; .001; 44.7% with \u226510-point improvement), and T\u00fcbingen CD-25 decreased by \u22127.8 points (P = .01). Early responses (by week 12) involved eating behaviour and depression symptoms. Delayed improvements (consolidating at weeks 24-36) were observed in sexual activity, environment, and social domains. Bodily restrictions (\u22124.3 points, P = .203) and cognition (\u22126.8 points, P = .143) did not show improvement over the study period. Item-level analysis identified a cluster of symptoms, predominantly psychological health anxiety and physical appearance concerns, where burden remained elevated throughout the 36 weeks despite cortisol normalization.<\/jats:p>\n                  <\/jats:sec>\n                  <jats:sec>\n                    <jats:title>Conclusions<\/jats:title>\n                    <jats:p>Metyrapone treatment is associated with meaningful, progressive HRQoL improvements across multiple symptom domains over 36 weeks, with a pattern consistent with rapid cortisol-mediated relief (eating, mood), delayed recovery in social and sexual domains, and persistent residual burden in symptom clusters likely driven by incomplete phenotypic reversal within the study timeframe.<\/jats:p>\n                  <\/jats:sec>","DOI":"10.1093\/ejendo\/lvag164","type":"journal-article","created":{"date-parts":[[2026,8,31]],"date-time":"2026-08-31T06:16:40Z","timestamp":1788157000000},"page":"431-442","source":"Crossref","is-referenced-by-count":1,"title":["Metyrapone-associated improvements in health-related quality of life in Cushing's syndrome"],"prefix":"10.1093","volume":"195","author":[{"given":"Emese","family":"Mez\u00f4si","sequence":"first","affiliation":[{"name":"Department of Internal Medicine, University Medical School , Pecs 7624 ,","place":["Hungary"]}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Lynnette K","family":"Nieman","sequence":"additional","affiliation":[{"name":"National Institute of Diabetes and Digestive and Kidney Diseases, National Institutes of Health , Bethesda, MD 20892 ,","place":["United 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26122 ,","place":["Germany"]}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Natacha","family":"Driessens","sequence":"additional","affiliation":[{"name":"Department of Endocrinology, H\u00f4pital Universitaire de Bruxelles H\u00f4pital Erasme, Universit\u00e9 Libre de Bruxelles , Brussels 1070 ,","place":["Belgium"]}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Carmen E","family":"Georgescu","sequence":"additional","affiliation":[{"name":"Department of Endocrinology, Iuliu Hatieganu University of Medicine and Pharmacy and Cluj County Emergency Hospital , Cluj-Napoca 400012 ,","place":["Romania"]}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Marcin","family":"Motyka","sequence":"additional","affiliation":[{"name":"Department of Endocrinology, Oncologic Endocrinology and Nuclear Medicine, University Hospital in Krakow , Krakow 30-688 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