Web Intervention for Parents of Youth With Genetic Syndromes (WINGS)

Browse trials for

• Tuberous Sclerosis 115
• Hamartoma Syndrome, Multiple 21
• Fragile X Syndrome 114
• Angelman Syndrome 52
• Rett Syndrome 96

Tuberous Sclerosis

115 studies on the registry are indexed under Tuberous Sclerosis; 27 are open to participants now.

This study's enrollment of 79 is above the median of 52 across 73 interventional studies indexed under Tuberous Sclerosis.

Lead sponsor

Rush University Medical Center is the lead sponsor of 397 studies on the registry; 61 are open to participants now.

Of its 32 completed or terminated interventional studies of FDA-regulated products, 26 (81%) have results posted.

Study arms

• Experimental
  Functional Behavioral Training

  Behavioral: Functional Behavioral Training (FBT)

• Active comparator
  Positive Parenting Strategies-Treatment As Usual

  Other: Positive Parenting Strategies-Treatment as Usual

Interventions

• BehavioralFunctional Behavioral Training (FBT)

  Function-based treatment (FBT) is a term for behavioral intervention approaches that use objective and systematic data collection to identify the cause of challenging behavior and teach a more prosocial replacement behavior. FBTslDD incorporates standard elements of FBT protocols with adaptations made specifically to support application to the syndromic IDD population. FBTslDD will include the following steps: (1) Syndromic IDD screening; (2) Stimulus preference assessment; (3) Indirect and descriptive assessments; (4) Screening for automatic function; (5) Functional analysis (FA); and (6) FBT intervention phase. If steps 1 - 5 determine a participant's challenging behavior serves a social function, Functional Communication Training (FCT) will be administered. If steps 1 - 5 determine a participant's challenging behavior serves an automatic function Competing Stimulus Treatment (CST) will be administered.

• OtherPositive Parenting Strategies-Treatment as Usual

  Using the Planned Adaptation approach, to identify proactive adaptations seeking to improve the fit of FBTsIDD with the unique needs of the syndromic IDD population. Triangulating mixed methods data from systematic video observations, questionnaires, and qualitative interviews, then the Framework for Reporting Adaptations and Modifications-Expanded24 (FRAME) will be used to develop a rich understanding of these and any additional adaptations made to the intervention when delivered by non-specialist providers within medical hubs serving syndromic IDD populations.

Primary outcomes

1. Clinical Global Impression

   Overall functioning will be measured using the Clinical Global Impression for Severity (CGI-S) and Improvement (CGl-I). GCI-S will be recorded at Baseline with CGI-S and CGI-I collected at all determined visits. The CGI provides a rating of the child's global functioning, taking into account all available information (e.g., child's symptoms, behavior, functioning). The CGI-S and CGl-I will be completed by a blinded independent evaluator.

   Time frame: TO (Baseline), T2 (Month 2), T4 (Month 4), T6 (Month 6)

Secondary outcomes

1. Parent Target Problems (PTP) Inventory

   The Parent Target Problems is a blinded rating provided by an independent evaluator and based on a caregiver interview (30-minutes at baseline; 10- to 15-minutes at follow-visits throughout the study). The independent evaluator will use the PTP interview to record the caregivers' estimate of the frequency of problematic behaviors. From this description, the independent evaluator (blind to treatment assignment) will generate a brief narrative describing the child's problem behaviors and their impact on daily functioning. The evaluator will read this back to the caregiver to provide corrections and/or add any pertinent missing information. This process will continue until the narrative represents an accurate description per caregiver report. Change on the chief concerns determined from interviews are then rated on a 9-point scale (1=normalized, 5=unchanged, 9=disastrous). The PTP has been used to measure outcomes in behavioral intervention trials.

   Time frame: TO (Baseline), T2 (Month 2), T4 (Month 4), T6 (Month 6)

2. Aberrant Behavior Checklist, Second Edition (ABC-2)

   The Aberrant Behavior Checklist, Second Edition (ABC-2) is a widely-used symptom checklist (completed by caregivers) that measures problematic behavior across five domain subscales: irritability, social withdrawal, stereotypic behavior, hyperactivity/noncompliance, inappropriate speech. Changes in raw scores across each subscale will be tracked against caregiver reports described in the PTP.

   Time frame: TO (Baseline), T2 (Month 2), T4 (Month 4), T6 (Month 6)

3. Adverse Event Monitoring

   Adverse events will be measured at each time point, and the log used in this study will be based on adverse event logs that have already been developed for other ongoing NIH trials in our center. The adverse event log will include data on any adverse events, the onset date, intensity of event(s), relationship to study (i.e., unrelated through definitely related), end date, outcome, regulatory action taken (if necessary), and whether treatment was required.

   Time frame: TO (Baseline), T1 (Month 1), T2 (Month 2), T3 (Month 3), T4 (Month 4), T5 (Month 5), T6 (Month 6)

Individual participant data

Plan to share: No