Marieke Johanna Kuiper1, Loïs Vrijenhoek1, Rick Brandsma1, Roelineke J Lunsing1, Huibert Burger2, Hendriekje Eggink1, Kathryn J Peall3, Maria Fiorella Contarino4,5, Johannes D Speelman4, Marina A J Tijssen1, Deborah A Sival6. 1. Department of Neurology University Medical Center Groningen, University of Groningen The Netherlands. 2. Department of General Practice University Medical Center Groningen, University of Groningen The Netherlands. 3. MRC Centre for Neuropsychiatric Genetics and Genomics Institute of Psychological Medicine and Clinical Neurosciences, School of Medicine, Cardiff University United Kingdom. 4. Department of Neurology Academic Medical Center, University of Amsterdam Amsterdam The Netherlands. 5. Department of Neurology Haga Teaching Hospital The Hague The Netherlands. 6. Department of Pediatrics Beatrix Children's Hospital University Medical Center Groningen, University of Groningen Groningen The Netherlands.
Abstract
BACKGROUND: The Burke-Fahn-Marsden Dystonia Rating Scale is a universally applied instrument for the quantitative assessment of dystonia in both children and adults. However, immature movements by healthy young children may also show "dystonic characteristics" as a consequence of physiologically incomplete brain maturation. This could implicate that Burke-Fahn-Marsden scale scores are confounded by pediatric age. OBJECTIVE: In healthy young children, we aimed to determine whether physiologically immature movements and postures can induce an age-related effect on Burke-Fahn-Marsden movement and disability scale scores. METHODS: Nine assessors specializied in movement disorders (3 adult neurologists, 3 pediatric neurologists, and 3 MD/PhD students) independently scored the Burke-Fahn-Marsden movement scale in 52 healthy children (4-16 years of age; 2 boys and 2 girls per year of age). Independent of that, parents scored their children's functional motor development according to the Burke-Fahn-Marsden disability scale in another 52 healthy children (4-16 years of age; 2 boys and 2 girls per year of age). By regression analysis, we determined the association between Burke-Fahn-Marsden movement and disability scales outcomes and pediatric age. RESULTS: In healthy children, assessment of physiologically immature motor performances by the Burke-Fahn-Marsden movement and disability scales showed an association between the outcomes of both scales and age (until 16 years and 12 years of age, β = -0.72 and β = -0.60, for Burke-Fahn-Marsden movement and disability scale, respectively [both P < 0.001]). CONCLUSIONS: The Burke-Fahn-Marsden movement and disability scales are influenced by the age of the child. For accurate interpretation of longitudinal Burke-Fahn-Marsden Dystonia Rating Scale scores in young dystonic children, consideration of pediatric age-relatedness appears advisory.
BACKGROUND: The Burke-Fahn-Marsden Dystonia Rating Scale is a universally applied instrument for the quantitative assessment of dystonia in both children and adults. However, immature movements by healthy young children may also show "dystonic characteristics" as a consequence of physiologically incomplete brain maturation. This could implicate that Burke-Fahn-Marsden scale scores are confounded by pediatric age. OBJECTIVE: In healthy young children, we aimed to determine whether physiologically immature movements and postures can induce an age-related effect on Burke-Fahn-Marsden movement and disability scale scores. METHODS: Nine assessors specializied in movement disorders (3 adult neurologists, 3 pediatric neurologists, and 3 MD/PhD students) independently scored the Burke-Fahn-Marsden movement scale in 52 healthy children (4-16 years of age; 2 boys and 2 girls per year of age). Independent of that, parents scored their children's functional motor development according to the Burke-Fahn-Marsden disability scale in another 52 healthy children (4-16 years of age; 2 boys and 2 girls per year of age). By regression analysis, we determined the association between Burke-Fahn-Marsden movement and disability scales outcomes and pediatric age. RESULTS: In healthy children, assessment of physiologically immature motor performances by the Burke-Fahn-Marsden movement and disability scales showed an association between the outcomes of both scales and age (until 16 years and 12 years of age, β = -0.72 and β = -0.60, for Burke-Fahn-Marsden movement and disability scale, respectively [both P < 0.001]). CONCLUSIONS: The Burke-Fahn-Marsden movement and disability scales are influenced by the age of the child. For accurate interpretation of longitudinal Burke-Fahn-Marsden Dystonia Rating Scale scores in young dystonic children, consideration of pediatric age-relatedness appears advisory.
Authors: S Durston; H E Hulshoff Pol; B J Casey; J N Giedd; J K Buitelaar; H van Engeland Journal: J Am Acad Child Adolesc Psychiatry Date: 2001-09 Impact factor: 8.829
Authors: E Monbaliu; E Ortibus; F Roelens; K Desloovere; J Deklerck; P Prinzie; P de Cock; H Feys Journal: Dev Med Child Neurol Date: 2010-01-28 Impact factor: 5.449
Authors: Nitin Gogtay; Jay N Giedd; Leslie Lusk; Kiralee M Hayashi; Deanna Greenstein; A Catherine Vaituzis; Tom F Nugent; David H Herman; Liv S Clasen; Arthur W Toga; Judith L Rapoport; Paul M Thompson Journal: Proc Natl Acad Sci U S A Date: 2004-05-17 Impact factor: 11.205
Authors: Deborah A Sival; Martinica Garofalo; Rick Brandsma; Tom A Bokkers; Marloes van den Berg; Tom J de Koning; Marina A J Tijssen; Dineke S Verbeek Journal: Diagnostics (Basel) Date: 2020-11-24