Feasibility, reliability, and clinical validity of the test of attentional performance for Children (KiTAP) in fragile X syndrome (FXS)

Andrew Knox, Andrea Schneider, Floridette Abucayan, Crystal Hervey, Christina Tran, David R Hessl, Elizabeth Berry-Kravis

Research output: Contribution to journalArticle

19 Citations (Scopus)

Abstract

Background: Attention and inhibition are core executive-function deficits in FRagile X syndrome (FXS). This pilot study evaluated the feasibility, reproducibility, and clinical relevance of the KiTAP, a computer-based pictorial measure of attention and inhibition with an enchanted-castle theme, in an FXS cohort. Methods: The 8-subtest KiTAP battery (as many subtests as each could perform) was given to 36 subjects with FXS, of variable age and cognitive/behavioral functioning, and 29 were retested, with an interval of 2 to 4 weeks between sessions. Subjects were rated by parents on the Aberrant Behavior Checklist-Community Edition (ABC-C) and Behavior Assessment System for Children, Second Edition (BASC-2). Feasibility, ceiling and basal effects, and data range and distribution analyses were used to eliminate outliers and invalid data points. Reproducibility of scores was analyzed using intraclass correlation coefficients (ICCs) and validity/clinical relevance was assessed by correlating KiTAP scores with ABC-C and BASC-2 scores. Results: Most of the participants with FXS were able to complete the Alertness, Distractibility, Flexibility, and Go/ NoGo subtests.About 50 to 60% completed the Visual Scanning and Vigilance subtests, and 20 to 25% completed the Sustained Attention and Divided Attention subtests. A panel of seven scores from four subtests were identified as feasible for most subjects, lacked excessive ceiling, basal, or learning effects, exhibited an acceptable range and distribution of scores, had good reproducibility (ICC > 0.7), and correlated with behavioral ratings for hyperactivity or attention (P < 0.01). Only minor differences in performance on the KiTAP were seen between mental agematched cohorts of subjects with FXS and non-FXS intellectual disability. Conclusions: The KiTAP can be administered to cohorts with FXS over a wide range of function with valid reproducible scores. With additional validation, it could represent a useful outcome measure for assessment of attention/executive-function abilities in clinical trials targeted to these core deficits in FXS.

Original languageEnglish (US)
Pages (from-to)1-12
Number of pages12
JournalJournal of Neurodevelopmental Disorders
Volume4
Issue number1
DOIs
StatePublished - 2012

Fingerprint

Fragile X Syndrome
Reproducibility of Results
Executive Function
Checklist
Outcome Assessment (Health Care)
Aptitude
Intellectual Disability
Parents
Clinical Trials
Learning

ASJC Scopus subject areas

  • Clinical Neurology
  • Pathology and Forensic Medicine
  • Cognitive Neuroscience
  • Pediatrics, Perinatology, and Child Health

Cite this

Feasibility, reliability, and clinical validity of the test of attentional performance for Children (KiTAP) in fragile X syndrome (FXS). / Knox, Andrew; Schneider, Andrea; Abucayan, Floridette; Hervey, Crystal; Tran, Christina; Hessl, David R; Berry-Kravis, Elizabeth.

In: Journal of Neurodevelopmental Disorders, Vol. 4, No. 1, 2012, p. 1-12.

Research output: Contribution to journalArticle

Knox, Andrew ; Schneider, Andrea ; Abucayan, Floridette ; Hervey, Crystal ; Tran, Christina ; Hessl, David R ; Berry-Kravis, Elizabeth. / Feasibility, reliability, and clinical validity of the test of attentional performance for Children (KiTAP) in fragile X syndrome (FXS). In: Journal of Neurodevelopmental Disorders. 2012 ; Vol. 4, No. 1. pp. 1-12.
@article{de0b180d72e7416b9b7f988a2cf4122b,
title = "Feasibility, reliability, and clinical validity of the test of attentional performance for Children (KiTAP) in fragile X syndrome (FXS)",
abstract = "Background: Attention and inhibition are core executive-function deficits in FRagile X syndrome (FXS). This pilot study evaluated the feasibility, reproducibility, and clinical relevance of the KiTAP, a computer-based pictorial measure of attention and inhibition with an enchanted-castle theme, in an FXS cohort. Methods: The 8-subtest KiTAP battery (as many subtests as each could perform) was given to 36 subjects with FXS, of variable age and cognitive/behavioral functioning, and 29 were retested, with an interval of 2 to 4 weeks between sessions. Subjects were rated by parents on the Aberrant Behavior Checklist-Community Edition (ABC-C) and Behavior Assessment System for Children, Second Edition (BASC-2). Feasibility, ceiling and basal effects, and data range and distribution analyses were used to eliminate outliers and invalid data points. Reproducibility of scores was analyzed using intraclass correlation coefficients (ICCs) and validity/clinical relevance was assessed by correlating KiTAP scores with ABC-C and BASC-2 scores. Results: Most of the participants with FXS were able to complete the Alertness, Distractibility, Flexibility, and Go/ NoGo subtests.About 50 to 60{\%} completed the Visual Scanning and Vigilance subtests, and 20 to 25{\%} completed the Sustained Attention and Divided Attention subtests. A panel of seven scores from four subtests were identified as feasible for most subjects, lacked excessive ceiling, basal, or learning effects, exhibited an acceptable range and distribution of scores, had good reproducibility (ICC > 0.7), and correlated with behavioral ratings for hyperactivity or attention (P < 0.01). Only minor differences in performance on the KiTAP were seen between mental agematched cohorts of subjects with FXS and non-FXS intellectual disability. Conclusions: The KiTAP can be administered to cohorts with FXS over a wide range of function with valid reproducible scores. With additional validation, it could represent a useful outcome measure for assessment of attention/executive-function abilities in clinical trials targeted to these core deficits in FXS.",
author = "Andrew Knox and Andrea Schneider and Floridette Abucayan and Crystal Hervey and Christina Tran and Hessl, {David R} and Elizabeth Berry-Kravis",
year = "2012",
doi = "10.1186/1866-1955-4-2",
language = "English (US)",
volume = "4",
pages = "1--12",
journal = "Journal of Neurodevelopmental Disorders",
issn = "1866-1947",
publisher = "Springer New York",
number = "1",

}

TY - JOUR

T1 - Feasibility, reliability, and clinical validity of the test of attentional performance for Children (KiTAP) in fragile X syndrome (FXS)

AU - Knox, Andrew

AU - Schneider, Andrea

AU - Abucayan, Floridette

AU - Hervey, Crystal

AU - Tran, Christina

AU - Hessl, David R

AU - Berry-Kravis, Elizabeth

PY - 2012

Y1 - 2012

N2 - Background: Attention and inhibition are core executive-function deficits in FRagile X syndrome (FXS). This pilot study evaluated the feasibility, reproducibility, and clinical relevance of the KiTAP, a computer-based pictorial measure of attention and inhibition with an enchanted-castle theme, in an FXS cohort. Methods: The 8-subtest KiTAP battery (as many subtests as each could perform) was given to 36 subjects with FXS, of variable age and cognitive/behavioral functioning, and 29 were retested, with an interval of 2 to 4 weeks between sessions. Subjects were rated by parents on the Aberrant Behavior Checklist-Community Edition (ABC-C) and Behavior Assessment System for Children, Second Edition (BASC-2). Feasibility, ceiling and basal effects, and data range and distribution analyses were used to eliminate outliers and invalid data points. Reproducibility of scores was analyzed using intraclass correlation coefficients (ICCs) and validity/clinical relevance was assessed by correlating KiTAP scores with ABC-C and BASC-2 scores. Results: Most of the participants with FXS were able to complete the Alertness, Distractibility, Flexibility, and Go/ NoGo subtests.About 50 to 60% completed the Visual Scanning and Vigilance subtests, and 20 to 25% completed the Sustained Attention and Divided Attention subtests. A panel of seven scores from four subtests were identified as feasible for most subjects, lacked excessive ceiling, basal, or learning effects, exhibited an acceptable range and distribution of scores, had good reproducibility (ICC > 0.7), and correlated with behavioral ratings for hyperactivity or attention (P < 0.01). Only minor differences in performance on the KiTAP were seen between mental agematched cohorts of subjects with FXS and non-FXS intellectual disability. Conclusions: The KiTAP can be administered to cohorts with FXS over a wide range of function with valid reproducible scores. With additional validation, it could represent a useful outcome measure for assessment of attention/executive-function abilities in clinical trials targeted to these core deficits in FXS.

AB - Background: Attention and inhibition are core executive-function deficits in FRagile X syndrome (FXS). This pilot study evaluated the feasibility, reproducibility, and clinical relevance of the KiTAP, a computer-based pictorial measure of attention and inhibition with an enchanted-castle theme, in an FXS cohort. Methods: The 8-subtest KiTAP battery (as many subtests as each could perform) was given to 36 subjects with FXS, of variable age and cognitive/behavioral functioning, and 29 were retested, with an interval of 2 to 4 weeks between sessions. Subjects were rated by parents on the Aberrant Behavior Checklist-Community Edition (ABC-C) and Behavior Assessment System for Children, Second Edition (BASC-2). Feasibility, ceiling and basal effects, and data range and distribution analyses were used to eliminate outliers and invalid data points. Reproducibility of scores was analyzed using intraclass correlation coefficients (ICCs) and validity/clinical relevance was assessed by correlating KiTAP scores with ABC-C and BASC-2 scores. Results: Most of the participants with FXS were able to complete the Alertness, Distractibility, Flexibility, and Go/ NoGo subtests.About 50 to 60% completed the Visual Scanning and Vigilance subtests, and 20 to 25% completed the Sustained Attention and Divided Attention subtests. A panel of seven scores from four subtests were identified as feasible for most subjects, lacked excessive ceiling, basal, or learning effects, exhibited an acceptable range and distribution of scores, had good reproducibility (ICC > 0.7), and correlated with behavioral ratings for hyperactivity or attention (P < 0.01). Only minor differences in performance on the KiTAP were seen between mental agematched cohorts of subjects with FXS and non-FXS intellectual disability. Conclusions: The KiTAP can be administered to cohorts with FXS over a wide range of function with valid reproducible scores. With additional validation, it could represent a useful outcome measure for assessment of attention/executive-function abilities in clinical trials targeted to these core deficits in FXS.

UR - http://www.scopus.com/inward/record.url?scp=84861859102&partnerID=8YFLogxK

UR - http://www.scopus.com/inward/citedby.url?scp=84861859102&partnerID=8YFLogxK

U2 - 10.1186/1866-1955-4-2

DO - 10.1186/1866-1955-4-2

M3 - Article

C2 - 22958782

AN - SCOPUS:84861859102

VL - 4

SP - 1

EP - 12

JO - Journal of Neurodevelopmental Disorders

JF - Journal of Neurodevelopmental Disorders

SN - 1866-1947

IS - 1

ER -