title: Feasibility and preliminary efficacy data from a computerized cognitive intervention in children with chromosome 22q11.2 deletion syndrome.
abstract: Children with chromosome 22q11.2 deletion syndrome (22q11DS) are significantly impaired in their academic performance and functionality due to cognitive deficits, especially in attention, memory, and other facets of executive function. Compounding these cognitive deficits is the remarkably high risk of major psychoses, occurring in 25% of adolescents and adults with the disorder. There are currently no evidence-based interventions designed to improve the cognitive deficits in these individuals. We implemented a neuroplasticity-based computerized cognitive remediation program for 12 weeks in 13 adolescents with 22q11DS, assessed feasibility, and measured changes in cognition before and after the intervention compared to a control group of 10 age- and gender-matched children with 22q11DS. Our results indicated that despite their cognitive impairments, this intervention is feasible in children with 22q11DS, with high rates of adherence and satisfaction. Our preliminary analyses indicate that gains in cognition occur with the intervention. Further study in a larger randomized controlled trial would enable assessment of efficacy of this novel intervention.
authors.affiliation: Division of Medical Genetics, Department of Pediatrics, Duke University Medical Center, Durham, NC, United States.
authors.forename: Waverly
authors.initials: W
authors.lastname: Harrell
authors.forename: Shaun
authors.initials: S
authors.lastname: Eack
authors.forename: Stephen R
authors.initials: SR
authors.lastname: Hooper
authors.forename: Matcheri S
authors.initials: MS
authors.lastname: Keshavan
authors.forename: Melanie S
authors.initials: MS
authors.lastname: Bonner
authors.forename: Kelly
authors.initials: K
authors.lastname: Schoch
authors.forename: Vandana
authors.initials: V
authors.lastname: Shashi
mesh_terms: D000015:Abnormalities, Multiple / Q000628:therapy*; D000293:Adolescent; D001288:Attention; D002648:Child; D002872:Chromosome Deletion; D002892:Chromosomes, Human, Pair 22; D003071:Cognition; D015928:Cognitive Behavioral Therapy / Q000379:methods* / Q000592:standards; D004062:DiGeorge Syndrome / Q000628:therapy*; D056344:Executive Function; D005240:Feasibility Studies; D005260:Female; D006801:Humans; D008297:Male; D008568:Memory; D009473:Neuronal Plasticity; D010865:Pilot Projects; D015203:Reproducibility of Results; D013813:Therapy, Computer-Assisted / Q000379:methods* / Q000592:standards; D016896:Treatment Outcome
keywords: 22q11DS; Chromosome 22q11.2 deletion syndrome; Cognitive remediation; DiGeorge syndrome; Schizophrenia; Velocardiofacial syndrome