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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.
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| Label | Type | Description | Intervention Names |
|---|---|---|---|
| Adaptive Cogmed | Experimental | 5 days per week for 5-6 weeks of parent aided visual working memory training, consisting of adaptive Cogmed |
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| Non-Adaptive Cogmed | Other | 5 days per week for 5-6 weeks of parent aided visual working memory training, consisting of non-adaptive Cogmed |
|
| Name | Type | Description | Arm Group Labels | Other Names |
|---|---|---|---|---|
| Working Memory Training | Other |
|
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| 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 |
| Measure | Description | 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) |
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Inclusion Criteria:
Exclusion Criteria:
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| Name | Affiliation | Role |
|---|---|---|
| David Hessl | UC Davis | Principal Investigator |
| Facility | Status | City | State | ZIP | Country | Contacts |
|---|---|---|---|---|---|---|
| M.I.N.D. Institute, U.C. Davis | Sacramento | California | 95817 | United States |
| PubMed Identifier | Type | Citation | Retractions |
|---|---|---|---|
| 30982467 | Derived | 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. |
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| ID | Term |
|---|---|
| D005600 | Fragile X Syndrome |
| ID | Term |
|---|---|
| D038901 | X-Linked Intellectual Disability |
| D008607 | Intellectual Disability |
| D019954 | Neurobehavioral Manifestations |
| D009461 | Neurologic Manifestations |
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| 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 |
| D009422 | Nervous System Diseases |
| D025064 | Sex Chromosome Disorders |
| D025063 | Chromosome Disorders |
| D000013 | Congenital Abnormalities |
| D009358 | Congenital, Hereditary, and Neonatal Diseases and Abnormalities |
| D030342 | Genetic Diseases, Inborn |
| D040181 | Genetic Diseases, X-Linked |
| D020271 | Heredodegenerative Disorders, Nervous System |