Treatment of Disruptive Behaviors in Fragile X Syndrome

NCT03510156 | Completed

• 1 other identifier: 36394
• Study Type: interventional
• Target: 60
• Locations: 1 country
• Sites: 1

Brief Summary

Disruptive behaviors such as self-injury, aggression, and property destruction pose significant health-related issues to children diagnosed with fragile X syndrome (FXS), impacting the child's quality of life and causing significant distress to families. Access to appropriate treatment for families is severely limited by factors such as cost of care, shortages of qualified treatment providers, and geographic spread of children with FXS across the country. To address these potential issues, the effectiveness of administering a standardized function-based behavioral treatment for problem behaviors in FXS will be evaluated using telemedicine. The proposed study intervention therefore offers a tremendous step forward in clinical research both in the field of FXS and in the field of developmental disabilities more broadly, and thus will have a significant impact on public health.

Conditions

Fragile X Syndrome; Disruptive Behavior

Outcome Measures

Primary Outcomes (1)

• Change from baseline level of problem behavior at 4, 8, 12 and 16 weeks

  Aberrant Behavior Checklist - Community (ABC-C)

  0, 4, 8, 12, 16 weeks

Secondary Outcomes (1)

• Change from baseline level of treatment acceptability at 4, 8, 12 and 16 weeks

  0, 4, 8, 12, 16 weeks

Study Arms (2)

Treatment

EXPERIMENTAL

Behavioral: Behavior analytic treatment

Observation

NO INTERVENTION

Interventions

Behavior analytic treatment BEHAVIORAL

Function-based treatment

Treatment

Eligibility Criteria

• Age: 3 Years - 10 Years
• Sex: male
• Healthy Volunteers: No
• Age Groups: Child (0-17)

You may qualify if:

• Child has a confirmed diagnosis of FXS (\>200 CGG repeats on the FMR1 gene with evidence of aberrant methylation)
• Child is male, between the ages of 3-10 years old
• Child is reported to show self-injury, property destruction and/or aggression on at least a daily basis
• The caregiver agrees to keep any therapies that the child receives (i.e., medications or other treatments) as stable as possible throughout involvement in the study
• The family has a high-speed internet connection at home or lives in an area with 4G network coverage
• Availability for one-hour daily telemedicine treatment sessions
• Availability for in-home assessment totaling 8 hours across two consecutive days

You may not qualify if:

• The child or caregiver has significant sensory impairments (e.g., blindness or deafness)
• Non-English speaking
• The child receives Applied Behavior Analysis services in excess of five hours per week
• The child has a significant neurological condition (e.g., frequent seizures, brain injury, Tourette's syndrome) that would preclude participation
• The child or caregiver has significant mobility issues
• The child is currently participating in another research study that would preclude participation in the study

Sponsors & Collaborators

• Stanford University: lead
• The John Merck Fund: collaborator

Study Sites (1)

Department of Psychiatry and Behavioral Sciences

Stanford, California, 94305, United States

Location

Related Publications (2)

• Hall SS, Rodriguez AB, Jo B, Pollard JS. Long-term follow-up of telehealth-enabled behavioral treatment for challenging behaviors in boys with fragile X syndrome. J Neurodev Disord. 2022 Sep 30;14(1):53. doi: 10.1186/s11689-022-09463-9.

  PMID: 36180840 DERIVED

• Hall SS, Monlux KD, Rodriguez AB, Jo B, Pollard JS. Telehealth-enabled behavioral treatment for problem behaviors in boys with fragile X syndrome: a randomized controlled trial. J Neurodev Disord. 2020 Nov 20;12(1):31. doi: 10.1186/s11689-020-09331-4.

  PMID: 33218305 DERIVED

MeSH Terms

Conditions

Fragile X Syndrome; Problem Behavior

Condition Hierarchy (Ancestors)

X-Linked Intellectual Disability; Intellectual Disability; Neurobehavioral Manifestations; Neurologic Manifestations; Nervous System Diseases; Sex Chromosome Disorders; Chromosome Disorders; Congenital Abnormalities; Congenital, Hereditary, and Neonatal Diseases and Abnormalities; Genetic Diseases, Inborn; Genetic Diseases, X-Linked; Heredodegenerative Disorders, Nervous System; Behavioral Symptoms; Behavior; Child Behavior

Study Officials

• Scott S Hall, PhD

  Stanford University

  PRINCIPAL INVESTIGATOR

Study Design

• Study Type: interventional
• Phase: not applicable
• Allocation: RANDOMIZED
• Masking: NONE
• Purpose: TREATMENT
• Intervention Model: PARALLEL
• Sponsor Type: OTHER
• Responsible Party: PRINCIPAL INVESTIGATOR
• PI Title: Associate Professor

Study Record Dates

• First Submitted: April 17, 2018
• First Posted: April 27, 2018
• Study Start: July 1, 2016
• Primary Completion: March 1, 2022
• Study Completion: March 1, 2022
• Last Updated: May 4, 2022

Record last verified: 2022-05

Data Sharing

• IPD Sharing: Will not share

Locations

US (1)