Fragile X Syndrome
Conditions
Brief summary
Individuals with fragile X syndrome (FXS) demonstrate profound executive function deficits that interfere with learning, socialization and emotion regulation. Extensive research focused on the animal models of FXS show that targeted pharmacological agents can normalize synaptic connectivity and reverse cognitive and behavioral deficits. This translational work has led to multiple national and international controlled trials in humans with FXS now underway. However, in contrast to the heavy focus on medication treatments, there have been no controlled trials to empirically-validate cognitive or behavioral interventions for FXS. The proposed study, the first non-pharmacological controlled trial for FXS, will evaluate the efficacy of Cogmed, a cognitive training program proven to enhance working memory and executive/frontal function in a variety of clinical populations. Demonstration of effective Cogmed training for FXS would represent a major advance in the field, one that may also generalize to other forms of intellectual disability. Furthermore, it is critical to determine whether the targeted pharmacological treatments can accelerate learning and cognitive development. Thus, the validation of Cogmed for FXS will provide a paradigm for testing hypotheses focused on combined efficacy of medication and cognitive training.
Interventions
OTHERWorking Memory Training
Sponsors
University of California, Davis
Study design
Allocation
RANDOMIZED
Intervention model
PARALLEL
Primary purpose
TREATMENT
Masking
QUADRUPLE (Subject, Caregiver, Investigator, Outcomes Assessor)
Eligibility
Sex/Gender
ALL
Age
8 Years to 18 Years
Healthy volunteers
No
Inclusion criteria
* fragile X full mutation * normal or corrected vision * English or Spanish speaking * ability to pass three-span items following completion of a Cogmed training session at baseline * parental agreement to maintain adherence to the training schedule and to not alter other treatments during the study unless medically necessary
Exclusion criteria
* previous Cogmed training * significant medical problems that would interfere with the study or significant brain trauma
Design outcomes
Primary
| Measure | Time frame | Description |
|---|---|---|
| Composite of Stanford Binet, Fifth Edition Block Span and Leiter-R Spatial Working Memory | 5-6 weeks | Average of z scores obtained from Block Span and Leiter-R Spatial Working Memory |
Secondary
| Measure | Time frame |
|---|---|
| Wechsler Intelligence Scale for Children-IV Digit Span | 5-6 weeks |
| Kiddie Test of Attentional Performance (KiTAP) | 5-6 weeks |
| Behavior Rating Inventory Rating of Executive Function (BRIEF; Teacher and Caregiver Reports) | 5-6 weeks |
| Swanson, Nolan and Pelham (SNAP-IV) | 5-6 weeks |
Countries
United States
Outcome results
None listed