Abstract

PURPOSE: Motor proficiency and physical activity (PA) levels are below average in youth with Prader-Willi syndrome (PWS), a rare neurodevelopmental disorder causing motor, behavioral, and medical challenges. This study aimed to determine if participation in a 24-week parent-led PA intervention led to sustained improvement in gross motor proficiency (MP) in youth with and without PWS. METHODS: Participants included 107 youth ages 8-16 with PWS or without PWS but categorized as obese, assigned to an intervention group or to a wait-list control group. After serving as controls, the wait-listed group received the intervention. Follow-up assessments were then conducted six months post intervention. The home-based PA program included playground and interactive console games scheduled 4 days a week. Training and program materials were provided to families at baseline to guide implementation of the program. Gross MP (Bruininks-Oseretsky Test of Motor Proficiency body coordination and strength and agility subtests) was obtained at baseline (pre), after 24 weeks of participating in the intervention (post) and at 6-month FU. Scale scores are reported (Mean ± SE). Intent-to-treat analyses were conducted. RESULTS: All youth demonstrated improved upper-limb coordination, bilateral coordination, balance, running speed and agility, and muscular strength at post (p<0.04 for all). At FU all youth maintained improvements in bilateral coordination (pre= 9.3±0.4, post=12.0±0.5, FU=11.6±0.6) and speed and agility (pre=9.2±0.4, post=10.8±, FU=11.4±0.5), p<0.05. At FU all youth maintained improvements in upper-limb coordination (pre=10.7±0.5, post=12.2±0.6, FU=12.1±0.8) balance (pre=8.1±0.3, post=9.3±0.4, FU=9.2±0.5) and strength (pre=8.0±0.3, post=9.0±0.4, FU=9.0±0.5), but the FU scores for these tests showed a slight decrease such that they were no longer significantly different from baseline (p<0.05). CONCLUSIONS:This parent-led game-based PA program resulted in immediate positive changes in gross MP in youth with and without PWS with improvements maintained for six months post intervention. Participation in a PA routine emphasizing motor skill development at home shows promise in leading to sustained improvements in MP in obese youth and in youth with PWS. Funded by USAMRAA W81XWH-11-1-0765

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