Family-based Intervention for Youth With Prader-Willi Syndrome: The Active Play at Home Study (APAH)

Family-based Exercise Intervention for Children and Adolescents With Prader-Willi Syndrome

Compared to other children, those with disability have additional challenges to being physically active. Prader-Willi Syndrome (PWS) is a genetic form of childhood obesity that is characterized by hypotonia, growth hormone deficiency, behavioral, and cognitive disability. In children, the low prevalence of PWS (1 in 10,000 to 15,000 live births) makes group-based physical activity (PA) interventions impossible. In contrast, the home environment presents a natural venue to establish a PA routine for this population. The present high prevalence of non-syndromal childhood obesity (one in four) and high physical inactivity rates, make alternative approaches to increasing PA in this population an area of high interest. Therefore, we have developed a 24-week home-based physical activity that could be suitable for children and adolescents ages 8-15 with PWS as well as obese children without PWS ages 8-11 years. It is hypothesized that: 1) an age-appropriate 24-week home-based PA intervention will increase PA levels in youth with PWS and without the syndrome but with obesity; 2) motor proficiency, central sensory reception and integration, and body composition will significantly improve in youth with and without PWS following completion of the home-based PA intervention and 3) physical activity self-efficacy and quality of life will increase significantly in youth with and without PWS who complete the home-based PA intervention. The study participants are 115 youth ages 8-15 y (45 with PWS and 70 without PWS but categorized as obese). The study utilizes a parallel design with the wait-listed control group receiving the intervention after serving as control. Participants are expected to complete the PA curriculum 4 days a week for six months including playground games 2 days a week and interactive console games 2 days a week. Parents are trained at baseline and then provided with a PA curriculum (Active Play at Home) and equipment to guide their implementation of the program at home. Measurements of children and parent dyads are assessed at baseline and at the end (week 24) of the intervention or control periods. Outcome measures include PA, body composition, motor proficiency, central sensory reception and integration (subsample of children only), quality of life and physical activity self-efficacy. PA intervention compliance is monitored using mail-in daily self-report checklists.

Study Overview

• Status: Unknown
• Conditions: Childhood Obesity; Prader Willi Syndrome
• Intervention / Treatment: Behavioral: Active Play at Home

Detailed Description

Compared to other children, those with disability have additional challenges to being physically active. Prader-Willi Syndrome (PWS) is a genetic form of childhood obesity that is characterized by hypotonia, growth hormone deficiency, behavioral, and cognitive disability. In children, the low prevalence of PWS (1 in 10,000 to 15,000 live births) makes group-based physical activity (PA) interventions impossible. In contrast, the home environment presents a natural venue to establish a PA routine for this population. The present high prevalence of non-syndromal childhood obesity (one in four) and high physical inactivity rates, make alternative approaches to increasing PA in this population an area of high interest. Specifically, approaches that involve the family have been identified as possible areas where further research is needed. Therefore, we have developed a 24-week home-based physical activity that could be suitable for children and adolescents ages 8-15 with PWS as well as obese children without PWS ages 8-11 years. It is hypothesized that: 1) an age-appropriate 24-week home-based PA intervention will increase PA levels in youth with PWS and without the syndrome but with obesity; 2) motor proficiency, central sensory reception and integration, and body composition will significantly improve in youth with and without PWS following completion of the home-based PA intervention and 3) physical activity self-efficacy and quality of life will increase significantly in youth with and without PWS who complete the home-based PA intervention. The study participants are 115 youth ages 8-15 y (45 with PWS and 70 without PWS but categorized as obese). The study utilizes a parallel design with the wait-listed control group receiving the intervention after serving as control. Participants are expected to complete the PA curriculum 4 days a week for six months including playground games 2 days a week and interactive console games 2 days a week. Parents are trained at baseline and then provided with a PA curriculum (Active Play at Home) and equipment to guide their implementation of the program at home. Tips related to scheduling and coping with barriers to daily program implementation are also included. Throughout, parents are contacted by phone once a week (weeks 1-4) and then every other week to provide support in between visits. Measurements of children and parent dyads are assessed at baseline, at 12-weeks of receiving the intervention and at the end (week 24) of the intervention or control periods. PA intervention compliance is monitored using mail-in daily self-report checklists.

• Study Type: Interventional
• Enrollment (Anticipated): 115
• Phase: Not Applicable

Contacts and Locations

Study Locations

• United States
  • California
    • Fullerton, California, United States, 92831
      • Recruiting
      • California State University Fullerton
      • Contact: Daniela A Rubin, Ph.D.; Phone Number: 4704 657-278-4704; Email: drubin@fullerton.edu
      • Contact: Diobel L Castner, M.S.; Phone Number: 8737 657-278-8737; Email: dcastner@fullerton.edu
      • Principal Investigator: Daniela A Rubin, Ph.D.
      • Sub-Investigator: Kathleen S Wilson, Ph.D.
      • Sub-Investigator: Debra J Rose, Ph.D.
      • Sub-Investigator: Leonard Wiersma, Ph.D.
  • Florida
    • Gainesville, Florida, United States, 32610
      • Recruiting
      • University of Florida Gainesville
      • Contact: Marilyn C Dumont-Driscoll, Ph.D. M.D.; Phone Number: 352-334-1340; Email: dumonmd@peds.ufl.edu
      • Principal Investigator: Marilyn C Dumont-Driscoll, Ph.D., M.D.
      • Sub-Investigator: Daniel Driscoll, Ph.D. M.D.

Participation Criteria

Eligibility Criteria

• Ages Eligible for Study: 4 years to 11 years (Child)
• Accepts Healthy Volunteers: No
• Genders Eligible for Study: All

Description

Inclusion Criteria:

• Having Prader-Willi Syndrome and being between ages 8-15 years. PWS status will be documented by appropriate molecular and cytogenetic testing (i.e., chromosomes, florescence in situ hybridization [FISH] 15, DNA methylation, DNA polymorphism studies)
• Being obese and between ages 8-11 years. Obesity is defined as having a body fat percentage greater than the 95th percentile (McCarthy, H. D., Cole, T. J., Fry, T., Jebb, S. A., & Prentice, A. M. (2006). Body fat reference curves for children. International Journal of Obesity (Lond), 30(4), 598-602).

Exclusion Criteria:

• Obese children without Prader-Willi Syndrome currently on lipid-lowering medication, diabetes medications, or blood pressure medications.
• Being pregnant

Study Plan

How is the study designed?

Design Details

• Primary Purpose: Treatment
• Allocation: Randomized
• Interventional Model: Parallel Assignment
• Masking: None (Open Label)

Number of Arms

2

Arms and Interventions

Participant Group / Arm	Intervention / Treatment
Experimental: Active Play at Home Intervention; Participant parents in the intervention arm will receive: 1) Active Play at Home curriculum and equipment, 2) Training session on Active Play at Home curriculum, 3) counseling on physical activity scheduling, identification of barriers, motivational strategies, 4) phone calls to check on compliance and issues with doing the program at home	Behavioral: Active Play at Home; The Active Play at Home (APAH) curriculum includes age-appropriate goal-oriented physical activities that combine playground and video games using the Nintendo Wii™ with exercises targeting: muscular strength and endurance, aerobic endurance, flexibility, balance, agility, and motor coordination. APAH was designed for children ages 8-11 without disability and ages 8-15 years with PWS. The playground games and interactive console-based games are to be performed twice weekly each. The activity is progressed from 25 to 45 minutes of moderate to vigorous physical activity throughout the 24 weeks period. Parents and children are trained to use the curriculum hands-on at baseline.
No Intervention: Wait-listed control; Participants will attend the baseline visit to do baseline measurements but will not receive any materials related to the Active Play at Home curriculum and will also not be contacted by phone during the control 24 weeks. After they serve as control group, they will be provided with the opportunity to receive the intervention.

What is the study measuring?

Primary Outcome Measures

Outcome Measure	Measure Description	Time Frame
Physical Activity	PA level: This outcome will be measured using accelerometers which provide detailed information on the temporal patterns (duration, frequency, and intensity) of PA. Data are stored as acceleration counts and data can be downloaded to a personal computer. Youth will use the 4MB GT3X (Actigraph, Pensacola, FL) triaxial activity monitor at the hip for eight consecutive days. Participants should wear the monitor all day while they are awake and remove the monitor when they shower, bathe, swim, or do something that may get the monitor completely wet. The youth and parents will be asked to fill out a log of all the physical activities they engage in during the days they wear the accelerometer. The child will wear the accelerometer for eight days to capture two typical weekend days. Physical activity will be defined as minutes per day of Moderate to Vigorous Physical Activity. Accelerometry cut-points published by Evenson et al. will be used to determine MVPA.	Baseline to 24 weeks

Secondary Outcome Measures

Outcome Measure	Measure Description	Time Frame
Body composition	Percentage of body fat will be measured using dual x-ray absorptiometry (DXA) model Lunar Prodigy Advance Plus (GE Healthcare, Milwaukee, WI). For female participants who have had their first menses, a urine pregnancy test will be completed before conducting the DXA scan. A pregnancy test is required by law because the x-rays might be harmful to the fetuses. If the participant is pregnant, she will be excluded from the study as a major study outcome is body composition and the participant will experience changes in body composition because of pregnancy, thus invalidating the study findings.	Baseline to 24 weeks
Motor proficiency	The Bruininks-Oseretsky Test of Motor Proficiency (BOTMP™-2) is used to evaluate overall motor proficiency. The BOTMP™-2 test measures fine manual control (fine motor precision and fine motor integration), manual coordination (manual dexterity and upper limb coordination), body coordination (bilateral coordination and balance), and strength and agility (running speed and agility and strength). This test also provides an overall motor proficiency score, as well as separate scores for the different domains of motor proficiency (Bruininks, R., Bruininks-Oseretsky test of motor proficiency: examiner's manual. 1978, MN: American Guidance Service). It is expected that significant improvements are demonstrated following completion of the PA intervention in the areas of upper limb coordination, bilateral coordination, balance, running speed, and agility and strength.	Baseline to 24 weeks
Sensory reception and motor integration	Sensory reception and motor integration will be measured using The Sensory Organization Test. The SOT is designed to identify impairments in one or more of the three sensory systems (i.e., vision, somatosensory, vestibular) that contribute to standing balance. This test has been previously used with pediatric populations, with and without disabilities such as cerebral palsy. The test is comprised of six test conditions. This test will be administered only in a subsample of participants (30 with PWS and 55 with non-syndromal obesity).	Baseline to 24 weeks
Physical activity self-efficacy	Self-efficacy for physical activity will be measured with an eight item questionnaire rated on a five point scale ranging from disagree a lot to agree a lot . This questionnaire was originally developed for use with children in fifth grade, but also validated with children in eighth grade. This questionnaire had a test-retest reliability of r= 0.84 over a period of two weeks. Additionally, the questionnaire had an internal consistency score of 0.88. Dishman, R.K., et al. Factorial invariance and latent mean structure of questionnaires measuring social-cognitive determinants of physical activity among black and white adolescent girls. Prev Med, 2002. 34(1): p. 100-8.	Baseline to 24 weeks
Quality of life	will be measured using Pediatric Quality of Life Inventory (PedsQL™) to assess multidimensional constructs covering physical, emotional, mental, social, and behavioral components of well-being and function in youth (Varni, J.W., et al., The PedsQL 4.0 as a pediatric population health measure: feasibility, reliability, and validity. Ambul Pediatr, 2003. 3(6): p. 329-41)	Baseline to 24 weeks

Other Outcome Measures

Outcome Measure	Measure Description	Time Frame
Anthropometrics	Stature, measured to the nearest 0.1 cm using a wall-mounted stadiometer. Body mass, obtained to the nearest 0.1 kg following Third U.S. National Health and Nutrition Examination Survey procedures. BMI will be computed by dividing body mass (kg) by stature (m2).	Baseline to 24 weeks
Dietary intake	To help interpret whether changes in body composition can be solely attributed to the PA intervention, dietary intake will also be assessed at the same time points as the other variables of interest. The participating parent or legal guardian will maintain a food record of the child's diet during two days of the week and one day on the weekend. In this record the parent will include quantity of food and fluids consumed, the preparation method, and the brand of the product. Before the baseline measurement, parents will attend a training session with a registered dietitian to learn how to estimate portion sizes and keep a food record. The information collected through the food records will be entered into The Food Processor, ESHA Research, Salem, OR, USA program and analyzed for macronutrient percent intake and total calories.	Baseline to 24 weeks
Parent confidence	Parent's Confidence: will be measured by the Proxy Self-efficacy questionnaire (Shields CA, & Brawley LR. (2006) Preferring proxy-agency: Impact on self-efficacy for exercise. Journal of Health Psychology,11, 904-914) to assess parents' confidence and self-efficacy in scheduling and managing their child's physical activity behaviors and adherence to the physical activity program.	Baseline to 24 weeks
Parent influence	Parent Influence: will be measured by the Parental Influence Question is Parental Social Control (Wilson, K.S., Spink, K.S., & Priebe, C.S. (2010). Parental social control in reaction to a hypothetical lapse in their child's activity: The role of parental activity and importance. Psychology of Sport and Exercise, 11, 231-237) to assess how the specific tactics parents choose to motivate their child influences the child's physical activity levels.	Baseline to 24 weeks

Collaborators and Investigators

• Sponsor: California State University, Fullerton
• Collaborators: University of Florida
• Investigators: Principal Investigator: Daniela A Rubin, Ph.D., California State University Fullerton, Department of Kinesiology

Publications and helpful links

General Publications

• Rubin DA, Wilson KS, Castner DM, Dumont-Driscoll MC. Changes in Health-Related Outcomes in Youth With Obesity in Response to a Home-Based Parent-Led Physical Activity Program. J Adolesc Health. 2019 Sep;65(3):323-330. doi: 10.1016/j.jadohealth.2018.11.014. Epub 2019 Mar 2.
• Rubin DA, Wilson KS, Dumont-Driscoll M, Rose DJ. Effectiveness of a Parent-led Physical Activity Intervention in Youth with Obesity. Med Sci Sports Exerc. 2019 Apr;51(4):805-813. doi: 10.1249/MSS.0000000000001835.
• Rubin DA, Wilson KS, Honea KE, Castner DM, McGarrah JG, Rose DJ, Dumont-Driscoll M. An evaluation of the implementation of a parent-led, games-based physical activity intervention: the Active Play at Home quasi-randomized trial. Health Educ Res. 2019 Feb 1;34(1):98-112. doi: 10.1093/her/cyy035.
• Rubin DA, Wilson KS, Wiersma LD, Weiss JW, Rose DJ. Rationale and design of active play @ home: a parent-led physical activity program for children with and without disability. BMC Pediatr. 2014 Feb 14;14:41. doi: 10.1186/1471-2431-14-41.

Study record dates

Study Major Dates

• Study Start: May 1, 2011
• Primary Completion (Anticipated): June 1, 2015
• Study Completion (Anticipated): October 1, 2015

Study Registration Dates

• First Submitted: February 6, 2014
• First Submitted That Met QC Criteria: February 7, 2014
• First Posted (Estimate): February 10, 2014

Study Record Updates

• Last Update Posted (Estimate): February 10, 2014
• Last Update Submitted That Met QC Criteria: February 7, 2014
• Last Verified: February 1, 2014

More Information

Terms related to this study

• Keywords: Family; Obesity; Childhood; Interactive games; Prader Willi syndrome; Playground games; Exercise routine
• Additional Relevant MeSH Terms: Pathologic Processes; Nervous System Diseases; Neurologic Manifestations; Neurobehavioral Manifestations; Disease; Congenital Abnormalities; Overnutrition; Nutrition Disorders; Overweight; Body Weight; Genetic Diseases, Inborn; Intellectual Disability; Abnormalities, Multiple; Chromosome Disorders; Obesity; Syndrome; Pediatric Obesity; Prader-Willi Syndrome
• Other Study ID Numbers: HSR-13-0142.54155; W81XWH-09-1-0682 (Other Grant/Funding Number: USAMRAA)