{"status":"ok","message-type":"work","message-version":"1.0.0","message":{"indexed":{"date-parts":[[2026,7,8]],"date-time":"2026-07-08T04:48:25Z","timestamp":1783486105732,"version":"3.55.0"},"reference-count":0,"publisher":"Oxford University Press (OUP)","issue":"3","content-domain":{"domain":["eje.bioscientifica.com"],"crossmark-restriction":true},"short-container-title":[],"published-print":{"date-parts":[[2020,3]]},"abstract":"<jats:sec>\n<jats:title>Objective<\/jats:title>\n<jats:p>To provide an overview of cognitive and motor outcome, and quality of life (QoL) in patients with congenital central hypothyroidism (CH-C).<\/jats:p>\n<\/jats:sec>\n<jats:sec>\n<jats:title>Design<\/jats:title>\n<jats:p>Systematic review with individual patient data (IPD) meta-analysis.<\/jats:p>\n<\/jats:sec>\n<jats:sec>\n<jats:title>Methods<\/jats:title>\n<jats:p>OVID MEDLINE, EMBASE and PsycInfo were searched from inception to June 11th, 2019. Studies in patients with CH-C, either isolated or with multiple pituitary hormone deficiency (MPHD), were included if CH-C patients could be separated from any additional patient groups. Primary outcomes were full-scale intelligence quotient (FSIQ) and motor outcome; secondary outcome was QoL. Following data-extraction, one-stage IPD meta-analysis was performed, fitting a linear mixed model with FSIQ as dependent variable. Random intercepts were fitted for each study.<\/jats:p>\n<\/jats:sec>\n<jats:sec>\n<jats:title>Results<\/jats:title>\n<jats:p>Six studies measuring FSIQ were eligible for meta-analysis, comprising 30 CH-C patients (20 males; 27 MPHD patients). FSIQ range was wide (64\u2013123). Mean weighted FSIQ was 97 (95% CI: 88\u2013105). Twenty-seven percent had an FSIQ below 85 (\u22651 <jats:sc>s.d.<\/jats:sc> below norm score), and 10% below 70 (\u22652 <jats:sc>s.d.<\/jats:sc> below norm score). There was no significant association between FSIQ and sex or age. Age at treatment initiation was available from three studies only, thus impeding a reliable analysis of this parameter. Motor outcome and QoL were each studied in one study; no quantitative analyses could be performed for these outcomes.<\/jats:p>\n<\/jats:sec>\n<jats:sec>\n<jats:title>Conclusion<\/jats:title>\n<jats:p>A wide range in FSIQ scores was observed in CH-C patients. Results should be interpreted with caution, because included patients mainly had MPHD and age at treatment initiation was unknown for the majority of patients.<\/jats:p>\n<\/jats:sec>","DOI":"10.1530\/eje-19-0874","type":"journal-article","created":{"date-parts":[[2020,1,21]],"date-time":"2020-01-21T16:57:09Z","timestamp":1579625829000},"page":"351-361","update-policy":"https:\/\/doi.org\/10.1530\/crossmarkpolicy-1","source":"Crossref","is-referenced-by-count":21,"title":["Cognitive outcome in congenital central hypothyroidism: a systematic review with meta-analysis of individual patient data"],"prefix":"10.1093","volume":"182","author":[{"given":"J C","family":"Naafs","sequence":"first","affiliation":[{"name":"1Department of Pediatric Endocrinology, Emma Children\u2019s Hospital, Amsterdam UMC, University of Amsterdam, Amsterdam, Netherlands"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"L M","family":"Vendrig","sequence":"additional","affiliation":[{"name":"1Department of Pediatric Endocrinology, Emma Children\u2019s Hospital, Amsterdam UMC, University of Amsterdam, Amsterdam, Netherlands"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"J","family":"Limpens","sequence":"additional","affiliation":[{"name":"2Medical Library, Amsterdam UMC, University of Amsterdam, Amsterdam, Netherlands"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"H J","family":"van der Lee","sequence":"additional","affiliation":[{"name":"3Pediatric Clinical Research Office, Amsterdam UMC, University of Amsterdam, Amsterdam, Netherlands"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"R G","family":"Duijnhoven","sequence":"additional","affiliation":[{"name":"4Department of Obstetrics and Gynaecology, Amsterdam UMC, University of Amsterdam, Amsterdam, Netherlands"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"J P","family":"Marchal","sequence":"additional","affiliation":[{"name":"5Pediatric Psychosocial Department, Emma Children\u2019s Hospital, Amsterdam UMC, University of Amsterdam, Amsterdam, Netherlands"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"A S van","family":"Trotsenburg","sequence":"additional","affiliation":[{"name":"1Department of Pediatric Endocrinology, Emma Children\u2019s Hospital, Amsterdam UMC, University of Amsterdam, Amsterdam, Netherlands"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"N","family":"Zwaveling-Soonawala","sequence":"additional","affiliation":[{"name":"1Department of Pediatric Endocrinology, Emma Children\u2019s Hospital, Amsterdam UMC, University of Amsterdam, Amsterdam, Netherlands"}],"role":[{"vocabulary":"crossref","role":"author"}]}],"member":"286","container-title":["European Journal of Endocrinology"],"original-title":[],"link":[{"URL":"https:\/\/eje.bioscientifica.com\/view\/journals\/eje\/182\/3\/EJE-19-0874.xml","content-type":"text\/html","content-version":"vor","intended-application":"text-mining"},{"URL":"https:\/\/eje.bioscientifica.com\/view\/journals\/eje\/182\/3\/EJE-19-0874.xml","content-type":"unspecified","content-version":"vor","intended-application":"similarity-checking"}],"deposited":{"date-parts":[[2023,2,14]],"date-time":"2023-02-14T16:14:10Z","timestamp":1676391250000},"score":1,"resource":{"primary":{"URL":"https:\/\/academic.oup.com\/ejendo\/article\/182\/3\/351\/6653944"}},"subtitle":[],"short-title":[],"issued":{"date-parts":[[2020,3]]},"references-count":0,"journal-issue":{"issue":"3"},"URL":"https:\/\/doi.org\/10.1530\/eje-19-0874","relation":{},"ISSN":["0804-4643","1479-683X"],"issn-type":[{"value":"0804-4643","type":"print"},{"value":"1479-683X","type":"electronic"}],"subject":[],"published":{"date-parts":[[2020,3]]}}}