Response Inhibition Training for Children With Williams Syndrome (WSRIT)

Developing Treatments to Improve Psychosocial Functioning in Children With Williams Syndrome Part 1: Response Inhibition Training for Children With Williams Syndrome

The investigators will conduct a pilot study investigating the effectiveness of a computerized response inhibition training program at reducing the response inhibition difficulties often seen in children with Williams syndrome ages 10-17. The investigators hypothesize that after completing the training program, children with Williams syndrome will show improvement on computerized measures of response inhibition and on parent measures of impulsivity.

Study Overview

• Status: Completed
• Conditions: Williams Syndrome
• Intervention / Treatment: Behavioral: Response Inhibition Training

Detailed Description

Behavioral characteristics of individuals with Williams syndrome include eagerness to approach and interact with others (including strangers), repeated questions especially about upcoming events, and difficulties with attention, impulsivity, and inhibition. There is very little systematic research about the kinds of interventions that are most useful for children with Williams syndrome to support optimal psychosocial functioning, and of the effectiveness of such interventions. We will conduct a pilot study of a potential intervention to address the response inhibition difficulties associated with Williams syndrome. The investigators will examine the utility of an online, web-based computerized cognitive retraining program aimed at addressing impulsivity and inhibition difficulties in 20 children, ages 10-17, with WS. The engaging training program has shown some utility in other populations, including children with tic disorders and trichotillomania. The investigators will examine the acceptability and feasibility of the treatment approach, as well as its impact on performance on experimental measures of impulsivity and on everyday psychosocial functioning as rated by parents; this study will consist of a small-scale pilot Randomized Clinical Trial with wait list. Through this novel work, we hope to develop and tune this approach for optimal outcomes and to demonstrate the promise of this intervention to address common challenges of people with Williams syndrome in the area of inhibition.

• Study Type: Interventional
• Enrollment (Actual): 25
• Phase: Not Applicable

Contacts and Locations

Study Locations

• United States
  • Wisconsin
    • Milwaukee, Wisconsin, United States, 53211
      • University of Wisconsin-Milwaukee Child Neurodevelopment Research Lab

Participation Criteria

Eligibility Criteria

• Ages Eligible for Study: 10 years to 17 years (CHILD)
• Accepts Healthy Volunteers: No
• Genders Eligible for Study: All

Description

Inclusion Criteria:

• Williams syndrome (diagnosed with genetic testing)
• Ages 10-17
• First language and main language spoken in the home is English
• Possess computer in the home that has internet access, and a second electronic device with access to Skype (e.g., smartphone, ipad, additional computer, ipod touch)

Exclusion Criteria:

• First language and main language spoken in the home is NOT English (because study measures and instructions are all in English)
• No computer in the home with internet access (because the study is being conducted via the internet)
• Comorbid severe illnesses or major surgery within the past 6 months
• Four or more previous sessions of inhibition training

Study Plan

How is the study designed?

Design Details

• Primary Purpose: TREATMENT
• Allocation: RANDOMIZED
• Interventional Model: CROSSOVER
• Masking: SINGLE

Number of Arms

2

Arms and Interventions

Participant Group / Arm	Intervention / Treatment
EXPERIMENTAL: Response Inhibition Training; Treatment group will receive immediate treatment after pre-test.	Behavioral: Response Inhibition Training; Computerized response inhibition training will be delivered via the Internet for 10 individual sessions.
NO_INTERVENTION: Waitlist Crossover; Waitlist group will not receive intervention while treatment group is active, but waitlist group will be offered treatment after post-test is completed.

What is the study measuring?

Primary Outcome Measures

Outcome Measure	Measure Description	Time Frame
Cognitive Inhibition Tasks	Three measures of response inhibition that were administered to children at pre-test will be administered after the 5-7-week training and again at a 3-month follow-up. Measures include computer-administered Motor Stroop task, Stop Signal task, and Go/No-Go Task.	5-7 weeks and 3 months

Secondary Outcome Measures

Outcome Measure	Measure Description	Time Frame
Conners-3	The Conners-3 will be administered to parents at post-intervention and again at follow-up to assess changes in attention and impulsivity.	5-7 weeks and 3 months
Emotion Regulation Checklist	The Emotion Regulation Checklist will be administered to parents at post-intervention and follow-up to assess changes in the child's ability to regulate emotions.	5-7 weeks and 3 months
Positive and Negative Affect Scale	The PANAS will be administered to parents at post-intervention and follow-up to assess changes in child positive and negative affect.	5-7 weeks and 3 months
Question-Asking Questionnaire	The QAQ will be administered to parents at post-intervention and follow-up to assess changes in amount and frequency of child anticipatory question asking.	5-7 weeks and 3 months
Spence Children's Anxiety Scale	The SCAS will be administered to parents at post-intervention and follow-up to assess changes in child anxieties.	5-7 weeks, 3 months
MINI Kid 6.0 Parent Version	The MINI Kid 6.0 Parent will be administered to parents at post-intervention and follow-up to assess changes in those areas endorsed at pre-intervention.	5-7 weeks and 3 months

Other Outcome Measures

Outcome Measure	Measure Description	Time Frame
Treatment Acceptability Questionnaire	Questionnaire about study acceptability will be administered at post test after the 5-7 week training.	5-7 weeks
Treatment Evaluation Inventory	Questionnaire evaluating treatment will be administered at post test after the 5-7 week training.	5-7 weeks

Collaborators and Investigators

• Sponsor: University of Wisconsin, Milwaukee
• Investigators: Principal Investigator: Bonita P Klein-Tasman, Ph.D., University of Wisconsin, Milwaukee

Study record dates

Study Major Dates

• Study Start: May 1, 2014
• Primary Completion (ACTUAL): May 1, 2016
• Study Completion (ACTUAL): May 1, 2016

Study Registration Dates

• First Submitted: August 6, 2014
• First Submitted That Met QC Criteria: August 7, 2014
• First Posted (ESTIMATE): August 8, 2014

Study Record Updates

• Last Update Posted (ACTUAL): May 31, 2017
• Last Update Submitted That Met QC Criteria: May 30, 2017
• Last Verified: May 1, 2017

More Information

Terms related to this study

• Keywords: Response Inhibition, Williams Syndrome
• Additional Relevant MeSH Terms: Pathologic Processes; Heart Diseases; Cardiovascular Diseases; Nervous System Diseases; Neurologic Manifestations; Neurobehavioral Manifestations; Disease; Congenital Abnormalities; Genetic Diseases, Inborn; Aortic Valve Disease; Heart Valve Diseases; Intellectual Disability; Chromosome Disorders; Aortic Valve Stenosis; Aortic Stenosis, Supravalvular; Syndrome; Williams Syndrome
• Other Study ID Numbers: 14.351