Motor proficiency in young children with Prader-Willi syndrome: a preliminary report

Authors

  • Daniela A. Rubin Department of Kinesiology, California State University Fullerton, 800 N. State College Blvd., Fullerton, CA 92831, USA - drubin@fullerton.edu https://orcid.org/0000-0003-1578-8251
  • Adam M. Hyde Department of Kinesiology, California State University Fullerton, 800 N. State College Blvd., Fullerton, CA 92831, USA https://orcid.org/0000-0002-9010-3219
  • Rachel M. Fenning Department of Child and Adolescent Studies and Center for Autism, California State University Fullerton, 800 N. State College Blvd., Fullerton, CA 92831, USA; Department of Psychological Science and The Claremont Autism Center, Claremont McKenna College, 888 Columbia Av., Claremont, CA 91711, USA https://orcid.org/0000-0003-4688-0499
  • Kathleen S. Wilson Department of Kinesiology, California State University Fullerton, 800 N. State College Blvd., Fullerton, CA 92831, USA https://orcid.org/0000-0001-7179-749X
  • Debra J. Rose https://orcid.org/0000-0001-7179-749X https://orcid.org/0000-0001-9845-9255

DOI:

https://doi.org/10.20338/bjmb.v18i1.373

Keywords:

Motor skills, Neurodevelopment, Obesity, Genetics, Prader-Willi syndrome

Abstract

BACKGROUND: Systematic documentation of motor characteristics in young children with Prader-Willi syndrome (PWS) is vital as access to treatments improves.

AIM: To characterize motor proficiency (MP) in young children with PWS.

METHOD: Participants included 6 children (3 male and 3 female) with PWS and 13 children with neurotypical development (NT), (9 male and 4 female) ages 4-6 years. Five out of six children with PWS had been on growth hormone replacement therapy (GHRT) for >3 years. Some children with PWS exhibited cognitive delays and others performed within the average range (Intellectual quotient mean± standard deviation = 65.3 ± 7.62, range = 47 – 94). MP was measured using the Short Form of the Bruininks-Oseretsky Test of Motor Proficiency-Second Edition (BOT-2-SF).

RESULTS: Children with PWS scored lower than children with NT in all areas of MP except for fine motor integration. All children with PWS scored well-below average for total MP; children with NT scored average (n=10) or above-average (n=3) for total MP, respectively.

CONCLUSION: At this young age children with PWS universally exhibited poor MP despite most of them being on GHRT and some exhibited intellectual functioning in the average range. Evidence of BOT-2-SF floor effects underscores the need to refine assessment procedures and enhance measurement precision for this population.

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Published

2024-04-27

How to Cite

Rubin, D. A., Hyde, A. M., Fenning, R. M., Wilson, K. S., & Rose, D. J. (2024). Motor proficiency in young children with Prader-Willi syndrome: a preliminary report . Brazilian Journal of Motor Behavior, 18(1). https://doi.org/10.20338/bjmb.v18i1.373

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Research Articles

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