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Research in developmental disabilities
Sep, 2013;34 (9): 2606-13.

Feasibility and preliminary efficacy data from a computerized cognitive intervention in children with chromosome 22q11.2 deletion syndrome.

Waverly Harrell, Shaun Eack, Stephen R Hooper, Matcheri S Keshavan, Melanie S Bonner, Kelly Schoch, Vandana Shashi
Author Information
  1. Waverly Harrell: Division of Medical Genetics, Department of Pediatrics, Duke University Medical Center, Durham, NC, United States.
PMID: 23751300 DOI: 10.1016/j.ridd.2013.05.009

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.

Keywords

22q11DS Chromosome 22q11.2 deletion syndrome Cognitive remediation DiGeorge syndrome Schizophrenia Velocardiofacial syndrome

References

  1. Genet Med. 2001 Jan-Feb;3(1):34-9 [PMID: 11339375]
  2. Schizophr Res. 2012 Aug;139(1-3):53-9 [PMID: 22695257]
  3. Front Hum Neurosci. 2012 Oct 02;6:271 [PMID: 23060775]
  4. Psychooncology. 2013 Aug;22(8):1856-65 [PMID: 23203754]
  5. Arch Gen Psychiatry. 1999 Oct;56(10):940-5 [PMID: 10530637]
  6. Arch Gen Psychiatry. 2010 Jul;67(7):674-82 [PMID: 20439824]
  7. J Child Psychol Psychiatry. 1993 Oct;34(7):1241-51 [PMID: 8245144]
  8. Res Dev Disabil. 2012 Jan-Feb;33(1):1-11 [PMID: 22093642]
  9. J Am Geriatr Soc. 2009 Apr;57(4):594-603 [PMID: 19220558]
  10. Asian J Psychiatr. 2011 Jun 1;4(2):119-124 [PMID: 21743818]
  11. Am J Psychiatry. 2009 Jun;166(6):639-41 [PMID: 19487398]
  12. Neuropsychology. 2008 Mar;22(2):226-34 [PMID: 18331165]
  13. Schizophr Res. 2006 Jun;84(2-3):187-93 [PMID: 16545541]
  14. J Pediatr. 2011 Apr;158(4):555-561.e4 [PMID: 21130467]
  15. Am J Psychiatry. 2011 May;168(5):472-85 [PMID: 21406461]
  16. Am J Psychiatry. 2013 Mar;170(3):275-89 [PMID: 23360949]
  17. Am J Psychiatry. 1996 Dec;153(12):1541-7 [PMID: 8942449]
  18. Schizophr Res. 2010 May;118(1-3):118-21 [PMID: 20056393]
  19. Am J Psychiatry. 2009 Jul;166(7):805-11 [PMID: 19448187]
  20. Am J Med Genet B Neuropsychiatr Genet. 2007 Jan 5;144B(1):27-36 [PMID: 17034021]
  21. Am J Med Genet. 1992 Jan 1;42(1):141-2 [PMID: 1308357]
  22. Br J Psychiatry. 2005 Feb;186:115-20 [PMID: 15684233]
  23. Schizophr Bull. 2010 Jul;36(4):869-79 [PMID: 19269924]
  24. Behav Genet. 2011 May;41(3):403-12 [PMID: 21573985]
  25. J Pediatr. 1999 Feb;134(2):193-8 [PMID: 9931529]
  26. Dev Disabil Res Rev. 2008;14(1):3-10 [PMID: 18636631]
  27. Dev Med Child Neurol. 2005 Dec;47(12):803-7 [PMID: 16288669]
  28. Schizophr Bull. 1998;24(1):163-74 [PMID: 9502554]
  29. Am J Hum Genet. 1992 May;50(5):924-33 [PMID: 1349199]
  30. Psychiatry Res. 1988 Nov;26(2):223-38 [PMID: 3237915]
  31. Schizophr Bull. 2009 Nov;35(6):1132-41 [PMID: 19745022]
  32. J Nerv Ment Dis. 1994 Aug;182(8):476-8 [PMID: 8040660]
  33. J Intellect Disabil Res. 2007 Sep;51(Pt 9):666-70 [PMID: 17845235]
  34. J Genet Couns. 2010 Oct;19(5):535-44 [PMID: 20680421]
  35. Curr Psychiatry Rep. 2009 Apr;11(2):99-105 [PMID: 19302762]
  36. Prog Brain Res. 2006;157:81-109 [PMID: 17046669]

Grants

  1. K23 MH095783/NIMH NIH HHS
  2. R01 MH078015/NIMH NIH HHS
  3. R34 MH091314/NIMH NIH HHS
  4. R34-MH091314/NIMH NIH HHS

MeSH Term

Abnormalities, Multiple
Adolescent
Attention
Child
Chromosome Deletion
Chromosomes, Human, Pair 22
Cognition
Cognitive Behavioral Therapy
DiGeorge Syndrome
Executive Function
Feasibility Studies
Female
Humans
Male
Memory
Neuronal Plasticity
Pilot Projects
Reproducibility of Results
Therapy, Computer-Assisted
Treatment Outcome
Controlled Clinical Trial Journal Article Research Support, N.I.H., Extramural Research Support, Non-U.S. Gov't
Article has an altmetric score of 3

Word Cloud

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