S T Lo1, P J L Collin2, A C S Hokken-Koelega3. 1. Department of Pediatric Endocrinology, Dutch Growth Research Foundation, Rotterdam, The Netherlands. 2. Department of Child Psychiatry, de Koraalgroep, Sittard, The Netherlands. 3. Department of Pediatrics, Subdivision of Endocrinology, Erasmus University Medical Center, Rotterdam, The Netherlands.
Abstract
BACKGROUND: Prader-Willi syndrome (PWS) is characterised by hypotonia, hypogonadism, short stature, obesity, behavioural problems, intellectual disability, and delay in language, social and motor development. There is very limited knowledge about visual-motor integration in children with PWS. METHOD: Seventy-three children with PWS aged 7-17 years were included. Visual-motor integration was assessed using the Beery Visual-motor Integration test at the start of the study and after 2 years. The association between visual-motor integration and age, gender, genetic subtype and intelligence was assessed. RESULTS: Children with PWS scored 'very low' (-3 standard deviations) in visual-motor integration and 'below average' (-1 standard deviation) in visual perception and motor coordination compared with typically developing children. Visual-motor integration was higher in children with a deletion (β = -0.170, P = 0.037), in older children (β = 0.222, P = 0.009) and in those with a higher total IQ (β = 0.784, P < 0.001). Visual perception was higher with a deletion (β = -0.193, P = 0.044) and higher IQ (β = -0.618, P < 0.001), but motor coordination was only higher with a higher total IQ (β = 0.429, P = 0.001). Visual perception and motor coordination were not associated with age or gender. There was a trend for visual-motor integration decline over the 2 year follow-up period (P = 0.099). Visual perception and motor coordination did not change over the follow-up period. CONCLUSIONS: Visual-motor integration is very poor in children with PWS. Children scored higher on the time-limited subtests for visual perception and motor coordination than the combined test for visual-motor integration. Separation of visual-motor integration tasks into pure visual or motor tasks and allowing sufficient time to perform the tasks might improve daily activities, both at home and at school.
BACKGROUND:Prader-Willi syndrome (PWS) is characterised by hypotonia, hypogonadism, short stature, obesity, behavioural problems, intellectual disability, and delay in language, social and motor development. There is very limited knowledge about visual-motor integration in children with PWS. METHOD: Seventy-three children with PWS aged 7-17 years were included. Visual-motor integration was assessed using the Beery Visual-motor Integration test at the start of the study and after 2 years. The association between visual-motor integration and age, gender, genetic subtype and intelligence was assessed. RESULTS:Children with PWS scored 'very low' (-3 standard deviations) in visual-motor integration and 'below average' (-1 standard deviation) in visual perception and motor coordination compared with typically developing children. Visual-motor integration was higher in children with a deletion (β = -0.170, P = 0.037), in older children (β = 0.222, P = 0.009) and in those with a higher total IQ (β = 0.784, P < 0.001). Visual perception was higher with a deletion (β = -0.193, P = 0.044) and higher IQ (β = -0.618, P < 0.001), but motor coordination was only higher with a higher total IQ (β = 0.429, P = 0.001). Visual perception and motor coordination were not associated with age or gender. There was a trend for visual-motor integration decline over the 2 year follow-up period (P = 0.099). Visual perception and motor coordination did not change over the follow-up period. CONCLUSIONS: Visual-motor integration is very poor in children with PWS. Children scored higher on the time-limited subtests for visual perception and motor coordination than the combined test for visual-motor integration. Separation of visual-motor integration tasks into pure visual or motor tasks and allowing sufficient time to perform the tasks might improve daily activities, both at home and at school.
Authors: Elisenda Bueichekú; Maite Aznárez-Sanado; Ibai Diez; Federico d'Oleire Uquillas; Laura Ortiz-Terán; Abid Y Qureshi; Maria Suñol; Silvia Basaia; Elena Ortiz-Terán; Maria A Pastor; Jorge Sepulcre Journal: Proc Natl Acad Sci U S A Date: 2020-03-06 Impact factor: 11.205