{"status":"ok","message-type":"work","message-version":"1.0.0","message":{"indexed":{"date-parts":[[2026,6,8]],"date-time":"2026-06-08T13:09:31Z","timestamp":1780924171311,"version":"3.54.1"},"reference-count":48,"publisher":"The Endocrine Society","issue":"10","license":[{"start":{"date-parts":[[2021,3,29]],"date-time":"2021-03-29T00:00:00Z","timestamp":1616976000000},"content-version":"vor","delay-in-days":0,"URL":"https:\/\/creativecommons.org\/licenses\/by-nc-nd\/4.0\/"}],"funder":[{"DOI":"10.13039\/100004319","name":"Pfizer","doi-asserted-by":"publisher","award":["WI219179"],"award-info":[{"award-number":["WI219179"]}],"id":[{"id":"10.13039\/100004319","id-type":"DOI","asserted-by":"publisher"}]}],"content-domain":{"domain":[],"crossmark-restriction":false},"short-container-title":[],"published-print":{"date-parts":[[2021,9,27]]},"abstract":"<jats:title>Abstract<\/jats:title>\n               <jats:sec>\n                  <jats:title>Context<\/jats:title>\n                  <jats:p>Central congenital hypothyroidism (CH) requires lifelong medical treatment. The majority of children with central CH have multiple pituitary hormone deficiencies (MPHD), but in some cases central CH is isolated. Most pituitary hormone deficiencies are associated with impaired health-related quality of life (HRQoL). However, studies on HRQoL in central CH are lacking.<\/jats:p>\n               <\/jats:sec>\n               <jats:sec>\n                  <jats:title>Objective<\/jats:title>\n                  <jats:p>To evaluate HRQoL and fatigue in children and young adults with central CH, as well as parent perspectives.<\/jats:p>\n               <\/jats:sec>\n               <jats:sec>\n                  <jats:title>Design<\/jats:title>\n                  <jats:p>Nationwide cross-sectional study comparing HRQoL between early-detected central CH patients and unaffected siblings with the Pediatric Quality of Life inventory (PedsQL\u2122) and PedsQL Multidimensional Fatigue Scale. Participants \u2265 8 years old filled in self-reports; parents of participants aged 3 to 18 years filled in parent reports. Isolated central CH patients, MPHD patients, and siblings were compared using a linear mixed model and Tukey\u2019s post hoc test.<\/jats:p>\n               <\/jats:sec>\n               <jats:sec>\n                  <jats:title>Results<\/jats:title>\n                  <jats:p>Eighty-eight patients and 52 siblings participated, yielding 98 self-reports and 115 parent reports. Isolated central CH patients (n\u2005=\u200535) and siblings showed similar scores on all subscales, both in the self-reports and parent reports. For MPHD patients (n\u2005=\u200553), self-reported scores were similar to those of siblings. Parent reported total HRQoL and fatigue scores were significantly poorer in MPHD patients compared with siblings (mean differences \u221210.2 and \u22129.4 points; P\u2005&amp;lt;\u20050.01), as were scores for physical functioning, social functioning and general fatigue.<\/jats:p>\n               <\/jats:sec>\n               <jats:sec>\n                  <jats:title>Conclusion<\/jats:title>\n                  <jats:p>Self-reported HRQoL scores in isolated central CH and MPHD patients were similar to siblings. However, parents reported significantly lower HRQoL and fatigue scores for MPHD patients, suggesting a difference in perceived limitations between MPHD patients and their parents.<\/jats:p>\n               <\/jats:sec>","DOI":"10.1210\/clinem\/dgab209","type":"journal-article","created":{"date-parts":[[2021,3,29]],"date-time":"2021-03-29T19:36:12Z","timestamp":1617046572000},"page":"e4231-e4241","source":"Crossref","is-referenced-by-count":7,"title":["Health-Related Quality of Life in Patients With Early-Detected Central Congenital Hypothyroidism"],"prefix":"10.1210","volume":"106","author":[{"ORCID":"https:\/\/orcid.org\/0000-0003-3787-0811","authenticated-orcid":false,"given":"Jolanda C","family":"Naafs","sequence":"first","affiliation":[{"name":"Emma Children\u2019s Hospital, Amsterdam UMC, University of Amsterdam, department of Pediatric Endocrinology, Amsterdam, the Netherlands"},{"name":"Amsterdam UMC, University of Amsterdam, department of Endocrinology and Metabolism, Amsterdam 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