{"status":"ok","message-type":"work","message-version":"1.0.0","message":{"indexed":{"date-parts":[[2026,10,29]],"date-time":"2026-10-29T01:27:40Z","timestamp":1793237260897,"version":"4.5.0"},"reference-count":25,"publisher":"Wiley","issue":"6","license":[{"start":{"date-parts":[[2020,1,9]],"date-time":"2020-01-09T00:00:00Z","timestamp":1578528000000},"content-version":"vor","delay-in-days":0,"URL":"http:\/\/onlinelibrary.wiley.com\/termsAndConditions#vor"}],"content-domain":{"domain":["onlinelibrary.wiley.com"],"crossmark-restriction":true},"short-container-title":["J Paediatrics Child Health"],"published-print":{"date-parts":[[2020,6]]},"abstract":"<jats:sec><jats:title>Aim<\/jats:title><jats:p>This study describes the prevalence and severity of perceived fatigue in a young neurofibromatosis type 1 (NF1) population.<\/jats:p><\/jats:sec><jats:sec><jats:title>Methods<\/jats:title><jats:p>Ethical approval was obtained and NF1 affected Individuals aged 2\u201318\u2009years from the Manchester's NF1 clinic invited along with any unaffected siblings. The PedsQL Multidimensional Fatigue Scale Parental and child report was used. This validated measure explores cognitive, physical and sleep\/rest domains on a 0\u2013100 scale. Higher scores indicate less fatigue. Fatigue scores in affected children were compared to unaffected siblings after adjusting for age, sex and Index of Multiple Deprivation and with published population standards using z\u2010scores.<\/jats:p><\/jats:sec><jats:sec><jats:title>Results<\/jats:title><jats:p>A total of 286 families were invited and 75 affected and 16 siblings participated. There were significant differences between NF1 and controls in the aggregated fatigue core (child report 55\u2009\u00b1\u200919 vs. 75 (14), <jats:styled-content style=\"fixed-case\"><jats:italic>P<\/jats:italic><\/jats:styled-content>\u2009&lt;\u20090.001; parent 54\u2009\u00b1\u200920 vs. 73\u2009\u00b1\u200918, <jats:styled-content style=\"fixed-case\"><jats:italic>P<\/jats:italic><\/jats:styled-content> = 0.001) and the three sub\u2010domains: cognitive (child 48\u2009\u00b1\u200927 vs. 75\u2009\u00b1\u200923, <jats:styled-content style=\"fixed-case\"><jats:italic>P<\/jats:italic><\/jats:styled-content>\u2009&lt;\u20090.001), physical (child 59\u2009\u00b1\u200919 vs. 82\u2009\u00b1\u200914, <jats:styled-content style=\"fixed-case\"><jats:italic>P<\/jats:italic><\/jats:styled-content>\u2009&lt;\u20090.001) and sleep\/rest (child 59\u2009\u00b1\u200919 vs. 71\u2009\u00b1\u200915, <jats:styled-content style=\"fixed-case\"><jats:italic>P<\/jats:italic><\/jats:styled-content> = 0.018). Similar differences were seen when compared with published controls (aggregated child <jats:styled-content style=\"fixed-case\"><jats:italic>z<\/jats:italic><\/jats:styled-content>\u2010score \u22121.9\u2009\u00b1\u20091.4, <jats:styled-content style=\"fixed-case\"><jats:italic>P<\/jats:italic><\/jats:styled-content>\u2009&lt;\u20090.001; parent \u22123.2\u2009\u00b1\u20091.8, <jats:styled-content style=\"fixed-case\"><jats:italic>P<\/jats:italic><\/jats:styled-content>\u2009&lt;\u20090.001). Prevalence of severe fatigue indicated by scores &lt;2 standard deviation below published means for healthy controls were also higher for children with NF on both parent and child reports. Agreement between child and parent reports were limited as is frequently seen in the literature.<\/jats:p><\/jats:sec><jats:sec><jats:title>Conclusion<\/jats:title><jats:p>This study suggests that children with NF1 are affected by perceived fatigue when compared with healthy children who do not have NF1.<\/jats:p><\/jats:sec>","DOI":"10.1111\/jpc.14764","type":"journal-article","created":{"date-parts":[[2020,1,9]],"date-time":"2020-01-09T12:00:10Z","timestamp":1578571210000},"page":"878-883","update-policy":"https:\/\/doi.org\/10.1002\/crossmark_policy","source":"Crossref","is-referenced-by-count":23,"title":["Perceived fatigue in children and young adults with neurofibromatosis type 1"],"prefix":"10.1111","volume":"56","author":[{"ORCID":"https:\/\/orcid.org\/0000-0001-6298-6600","authenticated-orcid":false,"given":"Grace","family":"Vassallo","sequence":"first","affiliation":[{"name":"Nationally Commissioned Complex NF1 Service Manchester University NHS Foundation Trust, Manchester Academic Health Sciences Centre  Manchester United Kingdom"},{"name":"NW Genomics Hub, Manchester Centre for Genomic Medicine, Division of Evolution and Genomic Sciences University of Manchester  Manchester United Kingdom"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Zulf","family":"Mughal","sequence":"additional","affiliation":[{"name":"Department of Paediatric Endocrinology and Bone Metabolism Manchester University NHS Foundation Trust, Manchester Academic Health Sciences Centre  Manchester United Kingdom"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Louise","family":"Robinson","sequence":"additional","affiliation":[{"name":"Nationally Commissioned Complex NF1 Service Manchester University NHS Foundation Trust, Manchester Academic Health Sciences Centre  Manchester United Kingdom"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Daniel","family":"Weisberg","sequence":"additional","affiliation":[{"name":"Nationally Commissioned Complex NF1 Service Manchester University NHS Foundation Trust, Manchester Academic Health Sciences Centre  Manchester United Kingdom"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Stephen A","family":"Roberts","sequence":"additional","affiliation":[{"name":"Centres for Biostatistics University of Manchester  Manchester United Kingdom"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Eileen","family":"Hupton","sequence":"additional","affiliation":[{"name":"Nationally Commissioned Complex NF1 Service Manchester University NHS Foundation Trust, Manchester Academic Health Sciences Centre  Manchester United Kingdom"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Judith","family":"Eelloo","sequence":"additional","affiliation":[{"name":"Nationally Commissioned Complex NF1 Service Manchester University NHS Foundation Trust, Manchester Academic Health Sciences Centre  Manchester United Kingdom"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Emma MM","family":"Burkitt Wright","sequence":"additional","affiliation":[{"name":"Nationally Commissioned Complex NF1 Service Manchester University NHS Foundation Trust, Manchester Academic Health Sciences Centre  Manchester United Kingdom"},{"name":"NW Genomics Hub, Manchester Centre for Genomic Medicine, Division of Evolution and Genomic Sciences University of Manchester  Manchester United Kingdom"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Shruti","family":"Garg","sequence":"additional","affiliation":[{"name":"Nationally Commissioned Complex NF1 Service Manchester University NHS Foundation Trust, Manchester Academic Health Sciences Centre  Manchester United Kingdom"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Lauren","family":"Lewis","sequence":"additional","affiliation":[{"name":"Nationally Commissioned Complex NF1 Service Manchester University NHS Foundation Trust, Manchester Academic Health Sciences Centre  Manchester United Kingdom"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"D Gareth","family":"Evans","sequence":"additional","affiliation":[{"name":"Nationally Commissioned Complex NF1 Service Manchester University NHS Foundation Trust, Manchester Academic Health Sciences Centre  Manchester United Kingdom"},{"name":"NW Genomics Hub, Manchester Centre for Genomic Medicine, Division of Evolution and Genomic Sciences University of Manchester  Manchester United Kingdom"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Stavros M","family":"Stivaros","sequence":"additional","affiliation":[{"name":"Nationally Commissioned Complex NF1 Service Manchester University NHS Foundation Trust, Manchester Academic Health Sciences Centre  Manchester United Kingdom"},{"name":"Academic Unit of Paediatric Radiology Royal Manchester Children's Hospital, Central Manchester University Hospitals NHS Foundation Trust, Manchester Academic Health Sciences Centre  Manchester United Kingdom"},{"name":"Division of Informatics, Imaging and Data Sciences, School of Health Sciences, Faculty of Biology, Medicine and Health University of Manchester, Manchester Academic Health Science Centre  Manchester United Kingdom"}],"role":[{"vocabulary":"crossref","role":"author"}]}],"member":"311","published-online":{"date-parts":[[2020,1,9]]},"reference":[{"key":"e_1_2_7_2_1","doi-asserted-by":"publisher","DOI":"10.1038\/gim.2017.215"},{"key":"e_1_2_7_3_1","doi-asserted-by":"publisher","DOI":"10.1002\/ajmg.a.33139"},{"key":"e_1_2_7_4_1","doi-asserted-by":"publisher","DOI":"10.1136\/jmg.2006.045906"},{"key":"e_1_2_7_5_1","doi-asserted-by":"publisher","DOI":"10.1212\/WNL.0b013e31827f07be"},{"key":"e_1_2_7_6_1","doi-asserted-by":"publisher","DOI":"10.1016\/j.nmd.2015.03.001"},{"key":"e_1_2_7_7_1","doi-asserted-by":"publisher","DOI":"10.1111\/j.1365-2214.2006.00717.x"},{"key":"e_1_2_7_8_1","doi-asserted-by":"publisher","DOI":"10.1111\/j.1365-2133.2008.08949.x"},{"key":"e_1_2_7_9_1","doi-asserted-by":"publisher","DOI":"10.1111\/dmcn.12361"},{"key":"e_1_2_7_10_1","first-page":"145","article-title":"Case\u2010control study of the muscular compartments and osseous strength in neurofibromatosis type 1 using 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