Promoting Prosocial Behavior in Syndromic Intellectual and Developmental Disabilities

Sponsor

Rush University Medical Center (Other)

Overall Status

Enrolling by invitation

CT.gov ID

NCT06139172

Collaborator

Emory University (Other)

Enrollment

Location

Arms

38.5

Anticipated Duration (Months)

2.4

Patients Per Site Per Month

Study Details

Study Description

Brief Summary

The purpose of this study is to evaluate the effectiveness of an adapted, telehealth functional behavioral therapy (FBTsIDD) specifically focused on promoting appropriate communication and behavioral strategies in individuals with syndromic intellectual and developmental disorders.

Participants will be asked to complete virtual study assessments at intake and then on a monthly basis for the duration of 3-6 months. In addition, participants will attend weekly or biweekly virtual intervention visits with a study therapist.

Condition or Disease	Intervention/Treatment	Phase
Telomeric 22Q13 Monosomy Syndrome Tuberous Sclerosis Hamartoma Syndrome, Multiple Fragile X Syndrome Angelman Syndrome Rett Syndrome Chromosome 15Q, Partial Deletion Creatine Deficiency, X-linked	Behavioral: Functional Behavioral Training (FBT) Other: Positive Parenting Strategies-Treatment as Usual	N/A

Study Design

Study Type:

Interventional

Anticipated Enrollment :

92 participants

Allocation:

Randomized

Intervention Model:

Parallel Assignment

Masking:

Single (Participant)

Primary Purpose:

Treatment

Official Title:

Promoting Prosocial Behavior in Syndromic Intellectual and Developmental Disabilities

Actual Study Start Date :

Sep 15, 2023

Anticipated Primary Completion Date :

Dec 1, 2025

Anticipated Study Completion Date :

Dec 1, 2026

Arms and Interventions

Arm	Intervention/Treatment
Experimental: Functional Behavioral Training	Behavioral: Functional 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.
Active Comparator: Positive Parenting Strategies-Treatment As Usual	Other: Positive 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.

Arm

Intervention/Treatment

Experimental: Functional Behavioral Training

Behavioral: Functional 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.

Active Comparator: Positive Parenting Strategies-Treatment As Usual

Other: Positive 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.

Outcome Measures

Primary Outcome Measures

Parent Target Problems (PTP) Inventory [TO (Baseline), T1 (Month 1), T2 (Month 2), T3 (Month 3), T4 (Month 4), T5 (Month 5), T6 (Month 6)]
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.

Secondary Outcome Measures

Aberrant Behavior Checklist, Second Edition (ABC-2) [TO (Baseline), T1 (Month 1), T2 (Month 2), T3 (Month 3), T4 (Month 4), T5 (Month 5), T6 (Month 6)]
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
Adverse Event Monitoring [TO (Baseline), T1 (Month 1), T2 (Month 2), T3 (Month 3), T4 (Month 4), T5 (Month 5), T6 (Month 6)]
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.
Clinical Global Impression [TO (Baseline), T1 (Month 1), T2 (Month 2), T3 (Month 3), T4 (Month 4), T5 (Month 5), T6 (Month 6)]
Overall functioning will be measured at each visit using the Clinical Global Impression for Severity (CGI-S) and Improvement (CGl-I). 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.

Eligibility Criteria

Criteria

Ages Eligible for Study:

2 Years to 12 Years

Sexes Eligible for Study:

All

Accepts Healthy Volunteers:

Inclusion Criteria:

Age(s) 2-12 years old at time of enrollment
Existing genetic syndrome based on clinical or genetic diagnosis and confirmed by medical records
Documented diagnosis of global developmental delay (GDD) or intellectual disability (ID)
estimated ID level in moderate, severe, or profound range
Disruptive behavior challenges determined to be clinically appropriate for remote, parent-implemented coaching based on clinician determination of acuity of problem behaviors
Caregiver who is able to consent in English.
Parent/caregiver available for weekly intervention sessions
Stable psychosocial and psychiatric treatments 3 months prior to baseline visit.

Exclusion Criteria:

High levels of aggression that mitigate remote or outpatient treatment as defined by clinician judgement and/or ABC Irritability scores above 20 (i.e., higher level of care needed than provided by study procedures)
Medical or psychiatric instability that may limit study participation
Meaningful change in medication or psychosocial interventions 3 months prior to baseline visit
Limitations in technology access that may hinder participation in remote trial (e.g., declining support provided by study participation)