Cognitive Training for Fragile X Syndrome

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.

Study Overview

• Status: Completed
• Conditions: Fragile X Syndrome
• Intervention / Treatment: Other: Working Memory Training

• Study Type: Interventional
• Enrollment (Actual): 106
• Phase: Not Applicable

Contacts and Locations

Study Locations

• United States
  • California
    • Sacramento, California, United States, 95817
      • M.I.N.D. Institute, U.C. Davis

Participation Criteria

Eligibility Criteria

• Ages Eligible for Study: 8 years to 18 years (Child, Adult)
• Accepts Healthy Volunteers: No
• Genders Eligible for Study: All

Description

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

Study Plan

How is the study designed?

Design Details

• Primary Purpose: Treatment
• Allocation: Randomized
• Interventional Model: Parallel Assignment
• Masking: Quadruple

Number of Arms

2

Arms and Interventions

Participant Group / Arm	Intervention / Treatment
Experimental: Adaptive Cogmed; 5 days per week for 5-6 weeks of parent aided visual working memory training, consisting of adaptive Cogmed	Other: Working Memory Training; Other Names: Cogmed
Other: Non-Adaptive Cogmed; 5 days per week for 5-6 weeks of parent aided visual working memory training, consisting of non-adaptive Cogmed	Other: Working Memory Training; Other Names: Cogmed

What is the study measuring?

Primary Outcome Measures

Outcome Measure	Measure Description	Time Frame
Composite of Stanford Binet, Fifth Edition Block Span and Leiter-R Spatial Working Memory	Average of z scores obtained from Block Span and Leiter-R Spatial Working Memory	5-6 weeks

Secondary Outcome Measures

Outcome Measure	Time Frame
Wechsler Intelligence Scale for Children-IV Digit Span	5-6 weeks
Wechsler Intelligence Scale for Children-IV Digit Span	3 month followup
Kiddie Test of Attentional Performance (KiTAP)	5-6 weeks
Kiddie Test of Attentional Performance (KiTAP)	3 month followup
Behavior Rating Inventory Rating of Executive Function (BRIEF; Teacher and Caregiver Reports)	5-6 weeks
Behavior Rating Inventory Rating of Executive Function (BRIEF; Teacher and Caregiver Reports)	3 month followup
Swanson, Nolan and Pelham (SNAP-IV)	5-6 weeks
Swanson, Nolan and Pelham (SNAP-IV)	3 month followup

Collaborators and Investigators

• Sponsor: University of California, Davis
• Investigators: Principal Investigator: David Hessl, UC Davis

Publications and helpful links

General Publications

• Hessl D, Schweitzer JB, Nguyen DV, McLennan YA, Johnston C, Shickman R, Chen Y. Cognitive training for children and adolescents with fragile X syndrome: a randomized controlled trial of Cogmed. J Neurodev Disord. 2019 Apr 15;11(1):4. doi: 10.1186/s11689-019-9264-2.

Study record dates

Study Major Dates

• Study Start (Actual): January 1, 2013
• Primary Completion (Actual): October 18, 2018
• Study Completion (Actual): October 18, 2018

Study Registration Dates

• First Submitted: April 5, 2016
• First Submitted That Met QC Criteria: April 18, 2016
• First Posted (Estimate): April 21, 2016

Study Record Updates

• Last Update Posted (Actual): May 3, 2019
• Last Update Submitted That Met QC Criteria: May 1, 2019
• Last Verified: May 1, 2019

More Information

Terms related to this study

• Additional Relevant MeSH Terms: Pathologic Processes; Nervous System Diseases; Neurologic Manifestations; Neurobehavioral Manifestations; Disease; Congenital Abnormalities; Genetic Diseases, Inborn; Genetic Diseases, X-Linked; Mental Retardation, X-Linked; Intellectual Disability; Heredodegenerative Disorders, Nervous System; Chromosome Disorders; Sex Chromosome Disorders; Syndrome; Fragile X Syndrome
• Other Study ID Numbers: 428005

Plan for Individual participant data (IPD)

• Plan to Share Individual Participant Data (IPD)?: Undecided