{"status":"ok","message-type":"work","message-version":"1.0.0","message":{"indexed":{"date-parts":[[2026,10,3]],"date-time":"2026-10-03T18:19:01Z","timestamp":1791051541307,"version":"4.1.0"},"reference-count":45,"publisher":"Wiley","issue":"5","license":[{"start":{"date-parts":[[2014,1,4]],"date-time":"2014-01-04T00:00:00Z","timestamp":1388793600000},"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":["Develop Med Child Neuro"],"published-print":{"date-parts":[[2014,5]]},"abstract":"<jats:sec><jats:title>Aim<\/jats:title><jats:p>Motor skill impairment and cognitive dysfunction are commonly reported features of neurofibromatosis type 1 (<jats:styled-content style=\"fixed-case\">NF<\/jats:styled-content>1). We characterized and determined the relationship between motor impairment, gait variables, and cognitive function in children and adolescents with <jats:styled-content style=\"fixed-case\">NF<\/jats:styled-content>1.<\/jats:p><\/jats:sec><jats:sec><jats:title>Method<\/jats:title><jats:p>Motor function, gait, and neurocognitive abilities were assessed in 46 children and adolescents with <jats:styled-content style=\"fixed-case\">NF<\/jats:styled-content>1 (26 males, 20 females; age range 7\u201317y; mean age 11y 1mo, <jats:styled-content style=\"fixed-case\">SD<\/jats:styled-content> 3y 2mo). Tests to establish correlations between neurocognitive, motor, and gait variables were performed.<\/jats:p><\/jats:sec><jats:sec><jats:title>Results<\/jats:title><jats:p>Compared with normative data, 28\/39 of our <jats:styled-content style=\"fixed-case\">NF<\/jats:styled-content>1 cohort demonstrated impaired performance for balance and upper limb coordination and 16\/38 for running speed and agility. Gait data revealed a strategy to preserve balance at the expense of velocity, with the unexpected exception of a tendency for reduced base of support. Neurocognitive testing confirmed mean <jats:styled-content style=\"fixed-case\">IQ<\/jats:styled-content> in the low average range (86.0) and deficits in spatial working memory and strategy generation. Significant correlations between a number of neurocognitive measures and motor abilities and gait were identified. The largest associations were between gait width and spatial working memory (<jats:italic>r<\/jats:italic>=0.594) and running speed and agility with strategy generation (<jats:italic>r<\/jats:italic>=0.549).<\/jats:p><\/jats:sec><jats:sec><jats:title>Interpretation<\/jats:title><jats:p>We have identified a relationship between balance, running speed and agility, gait, and cognition in children with <jats:styled-content style=\"fixed-case\">NF<\/jats:styled-content>1. Findings suggest a shared abnormal neurodevelopmental process underlying some cognitive and motor abilities in <jats:styled-content style=\"fixed-case\">NF<\/jats:styled-content>1. Results are discussed within the context of evidence highlighting abnormal dopamine\u2010mediated corticostriatal circuitry in <jats:styled-content style=\"fixed-case\">NF<\/jats:styled-content>1.<\/jats:p><\/jats:sec>","DOI":"10.1111\/dmcn.12361","type":"journal-article","created":{"date-parts":[[2014,1,4]],"date-time":"2014-01-04T10:32:52Z","timestamp":1388831572000},"page":"468-474","update-policy":"https:\/\/doi.org\/10.1002\/crossmark_policy","source":"Crossref","is-referenced-by-count":45,"title":["Relationship between cognitive dysfunction, gait, and motor impairment in children and adolescents with neurofibromatosis type 1"],"prefix":"10.1111","volume":"56","author":[{"given":"Joel A","family":"Champion","sequence":"first","affiliation":[{"name":"Discipline of Paediatrics and Child Health Faculty of Medicine University of Sydney Sydney NSW Australia"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Kristy J","family":"Rose","sequence":"additional","affiliation":[{"name":"Institute for Neuroscience and Muscle Research The Children's Hospital at Westmead Sydney NSW Australia"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Jonathan M","family":"Payne","sequence":"additional","affiliation":[{"name":"Discipline of Paediatrics and Child Health Faculty of Medicine University of Sydney Sydney NSW Australia"},{"name":"Institute for Neuroscience and Muscle Research The Children's Hospital at Westmead Sydney NSW Australia"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Joshua","family":"Burns","sequence":"additional","affiliation":[{"name":"Discipline of Paediatrics and Child Health Faculty of Medicine University of Sydney Sydney NSW Australia"},{"name":"Institute for Neuroscience and Muscle Research The Children's Hospital at Westmead Sydney NSW Australia"},{"name":"Arthritis and Musculoskeletal Research Group Faculty of Health Sciences University of Sydney Sydney NSW Australia"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Kathryn N","family":"North","sequence":"additional","affiliation":[{"name":"Discipline of Paediatrics and Child Health Faculty of Medicine University of Sydney Sydney NSW Australia"},{"name":"Institute for Neuroscience and Muscle Research The Children's Hospital at Westmead Sydney NSW Australia"},{"name":"Murdoch Childrens Research Institute The Royal Children's Hospital Melbourne Vic. 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