Autism & Developmental

Activity and participation in children with neurofibromatosis type 1.

Johnson et al. (2015) · Research in developmental disabilities 2015
★ The Verdict

Ten weeks of arm exercises helps NF1 kids move better for a moment, but it does not increase their everyday activity a year later.

✓ Read this if BCBAs writing participation goals for school-age kids with NF1 or similar motor delays.
✗ Skip if Clinicians only tracking strength gains, not life participation.

01Research in Context

01

What this study did

Researchers split kids with NF1 into two groups. One group did arm-strengthening games twice a week for ten weeks. The other group kept their usual routine.

After the ten weeks, kids kept exercising on their own for nine more months. The team checked arm skills and everyday participation at the start, ten weeks, and one year.

02

What they found

Right after training, the strengthening group moved their arms a little better. One year later both groups looked the same.

No group differences showed up in sports, hobbies, or classroom jobs at any point.

03

How this fits with other research

M-Symons et al. (2005) mapped the learning and visual problems common in NF1. Their list helps explain why short gym drills alone may not transfer to real life.

Gilboa et al. (2010) proved NF1 kids write slower and messier than peers. A strength program could help the arm but not the fine-motor planning those tasks need.

Lifshitz et al. (2014) showed kids with physical disabilities already join fewer play activities. Boudreau et al. (2015) now adds that ten weeks of strength work does not close that gap.

Hamama et al. (2021) found friendship is the toughest participation area for kids with ASD/ID. The same barrier likely blocks NF1 kids, and lifting weights does not touch social skills.

04

Why it matters

For NF1, brief muscle training gives a tiny skill bump that fades. To lift participation, pair any motor plan with social scripts, visual supports, and teacher training. Check the CLASS to spot which activities the child actually misses, then build those into goals.

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Add a peer buddy and a visual schedule to any motor task so strength practice turns into real play.

02At a glance

Intervention
other
Design
randomized controlled trial
Sample size
36
Population
developmental delay, other
Finding
mixed
Magnitude
small

03Original abstract

We describe activity and participation in children and youth with neurofibromatosis type 1 (NF1), and compared an intervention and control group after a strengthening program using the Pediatric Outcomes Data Collection Instrument (PODCI) and the Children's Assessment of Participation and Enjoyment (CAPE). Questionnaires were filled out by parents at baseline, 12-weeks, and 1-year. The intervention group performed a strengthening program twice a week for ten weeks, followed by a 9-month independent program. Thirty-six participants (18 control, 18 intervention) between the ages of 5- and 18-years (mean 10.6 years, SD 4.6 years) were enrolled, and 34 completed the 1-year assessment. There were significant differences between formal and informal participation (p<0.0001) in baseline CAPE scores for the entire cohort. At 12 weeks, PODCI upper extremity function improved in intervention and decreased in controls (p=0.040), while happiness declined in intervention and increased in control (p=0.003). There were no significant differences between control and intervention groups in any of the CAPE or PODCI change scores from baseline to 1-year. Upper extremity function, sport and physical function, comfort/pain and happiness PODCI scores were lower than normative values. The NF1 cohort had low participation in formal active physical and skill-based activities. The companionship and location dimensions suggest participation occurs with family and other relatives in the home or a relative's home and reflects a pattern of social isolation from peers.

Research in developmental disabilities, 2015 · doi:10.1016/j.ridd.2014.10.004