Cited by 6Williams Syndrome ResearchCognitive Abilities and TestingDevelopmental and Educational Neuropsychology

NIH Toolbox Cognition Battery Feasibility in Individuals With Williams Syndrome

Open access
Condy, Emma E., Becker, Lindsey, Farmer, Cristan, Kaat, Aaron J., Chlebowski, Colby, Kozel, Beth A., Thurm, Audrey·2022·American Journal on Intellectual and Developmental Disabilities

Abstract

The NIH Toolbox Cognition Battery (NIHTB-CB) was developed for epidemiological and longitudinal studies across a wide age span. Such a tool may be useful for intervention trials in conditions characterized by intellectual disability (ID), such as Williams syndrome (WS). Three NIHTB-CB tasks, including two executive functioning (Flanker, Dimensional Change Card Sort) and one episodic memory (Picture Sequence Memory) task, were given to 47 individuals with WS, ages 4 to 50, to evaluate feasibility (i.e., proportion of valid administrations) in this population. Findings indicated that NIHTB-CB tests showed good feasibility. Flanker and DCCS age-corrected scores were negatively correlated with age and showed floor effects, indicating these scores may not be useful for quantifying performance on these NIHTB-CB tests in ID.

AI snapshot

Written from the paper’s full text.

TL;DR

The NIH Toolbox Cognition Battery (NIHTB-CB) Flanker, DCCS, and Picture Sequence Memory Test (PSMT) were feasible to administer in 47 individuals with Williams syndrome ages 4–50, with 79% completing all three tasks and 89% completing each task individually. However, age-corrected scores on the executive function tasks showed floor effects and negative correlations with age, raising concerns about their validity for quantifying performance in intellectual disability.

Key findings

  • Feasibility was good: 79% (37/47) had valid administrations of all three NIHTB-CB tasks, and 89% completed each task individually (Flanker 40/45 after exclusions; DCCS 42/47; PSMT 42/47).
  • Age-corrected standard scores showed floor effects: 30% (12/40) of Flanker scores and 21% (9/42) of DCCS scores were at the floor (≤54), while no PSMT scores were at the floor.
  • Most participants scored at least two standard deviations below the mean on Flanker (83%; 33/40) and DCCS (76%; 32/42), but only 14% (6/42) did so on PSMT.
  • Age-corrected scores correlated negatively with age on Flanker (rs = −0.82, p < 0.01) and DCCS (rs = −0.45, p < 0.01), and NVIQ did not correlate with any NIHTB-CB task; Flanker and DCCS differed from PSMT in uncorrected scores (Friedman χ²(2) = 28.32, p < 0.001), but not from each other.

Methods

  • Participants were 47 individuals with Williams syndrome (ages 4–50; 25 males, 22 females; median FSIQ = 58, median NVIQ = 60; 80% with FSIQ <70), referred from an NIH natural history study and with molecular testing consistent with WS.
  • Three NIHTB-CB tasks were administered on iPad after IQ testing: Flanker (inhibitory control/attention), DCCS (cognitive flexibility), and PSMT (episodic memory), with standard procedures plus accommodations such as prompting/redirection as needed.
  • Analyses calculated proportion of valid administrations, examined floor effects in age-corrected standard scores, correlated scores with age and NVIQ, and compared performance across tasks using nonparametric repeated-measures ANOVA and follow-up Wilcoxon signed-rank tests in R 3.6.3.

Limitations

  • The paper states that using chronological age to select task versions may be a limitation because prior ID studies used mental age; it also notes limited administration in the 3–7-year-old versions and no data on co-occurring medical problems, psychiatric diagnoses, or medications.
  • The paper notes that accommodations such as prompting and redirecting could potentially affect Flanker and DCCS scoring because reaction time contributes to scores, though reaction time is only used when accuracy is ≥80%; it calls for further item-level investigation.
  • Evident from the text: the sample size is modest and the age range is wide, and the age-corrected score floor effect appears particularly problematic in older participants ages 20–50, limiting conclusions about score validity across the full age span.

Model-written summary. Check it against the paper before relying on it.

Integrity signals

Registry

Crossref · 10.1352/1944-7558-127.6.473

Cites

39 of 39 references verified0 citation errors · 0 not checkable

Statistics

Claims the paper makes

20 of 34 cited sources checked · 12 supported · 0 flagged · accuracy not yet measured

  • Premisecitation

    The lowest possible age-corrected standard score on the NIHTB-CB is 54 because scores were winsorized in a 2018 Toolbox update.

    “The lowest possible age-corrected standard score is 54 as they were winsorized (i.e., extreme values are limited through a transformation) in a Toolbox update in 2018, presumably because extreme scores have lower reliability.”
    Quote in the paperSubstantial claim1 of 1 sources checked
    • cites Shields et al., 2020 source

      The Methods (Data analysis) state that the current NIHTB-CB winsorizes age-corrected standard scores at 54, and the Discussion calls 54 the lower limit. Our copy does not date that change to a 2018 Toolbox update. The suggested reliability rationale is framed as the citing authors’ presumption, not a finding attributed to this source. (LLM, conf=0.88)

      Experimental — accuracy not yet measured
  • Interpretationcitation

    The discrepant performance between the PSMT and EF subtests appears consistent with previous findings using the NIHTB-CB in ID.

    “The discrepant performance between these subtests appears consistent with previous findings using the NIHTB-CB in ID (Hessl et al., 2016).”
    Quote in the paperNot a substantial claim1 of 1 sources checked
    • cites Hessl et al., 2016 source

      In Results, Study 3 (page not supplied), performance differed across diagnostic groups on the flanker executive-function test but not on the picture sequence memory task (PSMT). That supports a discrepancy between PSMT and flanker, but the paper found no corresponding group difference on DCCS, the other executive-function subtest, so it does not establish the discrepancy for EF subtests collectively. (LLM, conf=0.83)

      Experimental — accuracy not yet measured
  • Premisecitation

    Previous studies examining the NIHTB-CB in ID used mental age to determine which subtest version was administered.

    “previous studies examining the NIHTB-CB in ID used mental age to determine which subtest version was administered (Hessl et al., 2016; Shields et al., 2020).”
    Quote in the paperSubstantial claim2 of 2 sources checked
    • cites Hessl et al., 2016 source
    • cites Shields et al., 2020 source
  • Premisecitation

    In individuals with ID, age-corrected standard scores often decrease over time even though their ability may have actually improved, due to slower rate of increasing cognitive skills compared to same-aged peers.

    “due to the slower rate of increasing cognitive skills in people with ID, compared to their same-aged peers, age-corrected standard scores often decrease over time in individuals with ID even though their ability may have actually improved (Farmer et al., 2020).”
    Quote in the paperSubstantial claim1 of 1 sources checked
    • cites Farmer et al., 2020 source

      The Introduction explains that when children with neurodevelopmental disorders gain ability more slowly than their age peers, age-normed scores can obscure improvement and may even decline. It does not establish that scores often decline in individuals with intellectual disability specifically; the Results’ Differential Ability Scale-II illustration instead reports stable or modestly improved norm-referenced scores. (LLM, conf=0.86)

      Experimental — accuracy not yet measured
  • Findingown data

    The PSMT task was completed with valid scores by 89% (42/47) of individuals with Williams syndrome who attempted it.

    “The NIHTB-CB PSMT subtest was attempted with 47 participants with WS. Of these, 42 (89%) were able to complete the task and receive scores through the NIHTB-CB”
    Quote in the paperSubstantial claim
  • May combine two claims. 22
  • Classified as probably not a substantial claim. 21
  • The quote only partly states this claim. 1
  • May combine two claims.

    Premisecitation

    A particular challenge for measuring change in intellectual disability is the potential for floor effects, which may lead to a mischaracterization of a potentially meaningful change over time in individuals with intellectual disability.

    “However, a particular challenge for measuring change in ID is the potential for floor effects (Hessl et al., 2009; Sansone et al., 2014), which may lead to a mischaracterization of a potentially meaningful change over time in individuals with ID (Bishop et al., 2015; Thurm et al., 2020).”
    Quote in the paperSubstantial claim2 of 3 sources checked
    • cites Hessl et al., 2009 source
    • cites Sansone et al., 2014 source
    • cites Bishop et al., 2015

      Couldn’t be matched to a paper in Crossref, so it can’t be checked.

  • Probably not a substantial claim: may not pass S3 (load-bearing). May combine two claims.

    Premisecitation

    The NIHTB-CB was normed in a demographically diverse sample of 4,859 participants ages 3-85, with eligibility criteria including the capability of following test instructions and adequate visual and auditory functioning.

    “The battery was normed in a demographically diverse sample of 4,859 participants ages 3–85, with eligibility criteria including the capability of following test instructions and adequate visual and auditory functioning (Beaumont et al., 2013).”
    Quote in the paperNot a substantial claim1 of 1 sources checked
    • cites Beaumont et al., 2013 source

      The Abstract describes a planned norming sample ages 3–85, stratified by demographic characteristics, with at least 4,205 participants—not a completed sample of 4,859. RELEVANT POPULATION lists the ability to follow test instructions as an eligibility criterion, but says adequate visual and auditory functioning is required only for a subset of measures. Page numbers are not available in our copy. (LLM, conf=0.90)

      Experimental — accuracy not yet measured
  • May combine two claims.

    Interpretationcitation

    The rates of successful administration for each NIHTB-CB task in Williams syndrome are comparable to the initial validation study of the Toolbox in typically developing children and to those previously reported in individuals with intellectual disability, though higher on the DCCS in the present study (90% vs. 64% in the previous study of individuals with ID).

    “These rates of successful administrations for each task are comparable to the initial validation study of the Toolbox in typically developing children and to those previously reported in individuals with ID (Shields et al., 2020), though higher on the DCCS in the present study (90% vs. 64% in the previous study of individuals with ID).”
    Quote in the paperSubstantial claim1 of 1 sources checked
    • cites Shields et al., 2020 source

      Split into 2 atoms — 0 confirmed, 2 partially supported, 0 disputed, 0 misattributed, 0 unverifiable. Expand the claim to see per-atom verdicts.

      Experimental — accuracy not yet measured
  • Probably not a substantial claim: may not pass S3 (load-bearing). May combine two claims.

    Premisecitation

    The NIHTB-CB consists of seven tasks measuring different domains of cognitive function, including executive functioning, memory, processing speed, and language.

    “The NIH Toolbox Cognition Battery (NIHTB-CB) consists of seven tasks measuring different domains of cognitive function, including EF, memory, processing speed, and language (Weintraub et al., 2014; Zelazo et al., 2013).”
    Quote in the paperNot a substantial claim1 of 2 sources checked
    • cites Weintraub et al., 2014 source
    • cites Zelazo et al., 2013 source
  • Probably not a substantial claim: may not pass S3 (load-bearing).

    Premisecitation

    Attentional issues are widely reported in WS.

    “which is unsurprising given the attentional issues widely reported in WS (Breckenridge et al., 2013; Greer et al., 2017; Leyfer et al., 2006).”
    Quote in the paperNot a substantial claim2 of 3 sources checked
    • cites Breckenridge et al., 2013

      Couldn’t be matched to a paper in Crossref, so it can’t be checked.

    • cites Greer et al., 2017 source
    • cites Leyfer et al., 2006 source
  • May combine two claims.

    Premisecitation

    Efforts to validate the NIHTB-CB in individuals with intellectual disability indicate good feasibility in the moderate ID range and a mental age above 5 years and convergent validity comparable to the original NIHTB-CB norming study.

    “Efforts to validate the measure in individuals with ID indicate good feasibility in the moderate ID range and a mental age above 5 years (Shields et al., 2020) and convergent validity comparable to the original NIHTB-CB norming study (Hessl et al., 2016).”
    Quote in the paperSubstantial claim2 of 2 sources checked
    • cites Shields et al., 2020 source
    • cites Hessl et al., 2016 source
  • May combine two claims.

    Premisecitation

    The present study based the task version administered on chronological age, unlike other studies looking at the Toolbox in ID, which have used mental age as estimated through IQ testing.

    “The present study based the task version administered on chronological age, unlike other studies looking at the Toolbox in ID, which have used mental age as estimated through IQ testing.”
    Quote in the paperNot a substantial claim2 of 2 sources checked
    • cites Hessl et al., 2016 source

      The Results (Study 3, Table 6 note; page not available) say that mental age was measured by Stanford-Binet 5 IQ testing. But the paper does not say mental age determined which Toolbox task version was administered; it also says mental age in Study 2 was estimated from chart review and examiner judgment, not IQ testing. (LLM, conf=0.82)

      Experimental — accuracy not yet measured
    • cites Shields et al., 2020 source
  • Probably not a substantial claim: may not pass S3 (load-bearing).

    Premisecitation

    Previous studies using the NIHTB-CB in ID have used a z-score deviation scoring approach.

    “Alternatively, previous studies using the NIHTB-CB in ID have used a z-score deviation scoring approach (Hessl et al., 2009; Shields et al., 2020).”
    Quote in the paperNot a substantial claim2 of 2 sources checked
    • cites Hessl et al., 2009 source

      The Methods (Statistical methods; page unavailable) describe calculating age-based z-score deviations for children with intellectual disabilities. But the Methods (Measures; page unavailable) identify the test as the WISC-III, not the NIHTB-CB. This source supports the scoring approach, but not the claim that it was used with the NIHTB-CB. (LLM, conf=0.89)

      Experimental — accuracy not yet measured
    • cites Shields et al., 2020 source
  • Probably not a substantial claim: may not pass S3 (load-bearing). May combine two claims.

    Premisecitation

    The NIHTB-CB provides uncorrected standard scores that measure absolute performance and are argued to be a more appropriate metric in intellectual disability.

    “However, the NIHTB-CB also provides uncorrected standard scores, serving as a metric more akin to a person ability score, that measure absolute performance and are argued to be a more appropriate metric in ID (Farmer et al., 2020).”
    Quote in the paperNot a substantial claim1 of 1 sources checked
    • cites Farmer et al., 2020 source

      The Introduction (page unavailable) distinguishes person ability scores, which quantify ability, from norm-referenced standard scores, which do not convey absolute ability. The Discussion (page unavailable) argues that ability scores can be a useful alternative for studies of neurodevelopmental disorders. The paper does not establish that the NIHTB-CB provides uncorrected standard scores or that those particular scores measure absolute performance. (LLM, conf=0.85)

      Experimental — accuracy not yet measured
  • May combine two claims. The quote only partly states this claim.

    Premisecitation

    A floor effect was noted for age-corrected standard scoring of the NIHTB-CB measures in intellectual disability, which was remedied through a rescoring procedure.

    “A floor effect was noted for age-corrected standard scoring of the measures, which was remedied through a rescoring procedure.”
    Quote in the paperSubstantial claim1 of 1 sources checked
    • cites Shields et al., 2020 source
  • May combine two claims.

    Findingown data

    The difference in uncorrected standard scores across NIHTB-CB tests in Williams syndrome was driven by differences between the Flanker and PSMT and between the DCCS and PSMT, while Flanker and DCCS scores did not differ.

    “Follow-up Wilcox Signed-Rank tests indicated this difference was driven by differences between the Flanker (Mdn = 65, IQR = 57–68) and PSMT (Mdn = 82, IQR = 79–88), (V = 19, p < 0.001) and DCCS (Mdn = 56, IQR = 51–78) and PSMT (V = 55, p < 0.001); however, the Flanker and DCCS scores did not differ (V = 299.5, p = 0.98).”
    Quote in the paperNot a substantial claim
  • May combine two claims.

    Findingown data

    Most individuals with Williams syndrome who completed the tasks had an age-corrected standard score at least two standard deviations below the mean (≤70) on the Flanker (83%; 33/40) and DCCS (76%; 32/42), while only 14% (6/42) scored in this range on the PSMT.

    “Most individuals with WS who completed the tasks had an age-corrected SS that was at least two standard deviations below the mean (≤ 70) on the Flanker (83%; 33/40) and DCCS (76%; 32/42); only 14% (6/42) scored in this lower range on the PSMT.”
    Quote in the paperSubstantial claim
    • Rests on an assumption: The NIHTB-CB tasks measure the same cognitive constructs across their different age versions despite differences in stimuli, instructions, and prompts.
  • May combine two claims.

    Findingown data

    In individuals with Williams syndrome ages 4 to 50, the NIH Toolbox Cognition Battery Flanker, Dimensional Change Card Sort, and Picture Sequence Memory tasks showed good feasibility, with 89% able to complete each task and 79% able to complete all three.

    “Overall, the NIHTB-CB tasks showed good feasibility for use in people with WS, with 89% able to complete each task, and the majority able to complete all three tasks (79%).”
    Quote in the paperSubstantial claim
  • May combine two claims.

    Findingown data

    No individuals with Williams syndrome scored at the floor on the PSMT, whereas 30% (12/40) of Flanker scores and 21% (9/42) of DCCS scores were at the floor.

    “No WS participants scored at the floor on the PSMT; however, 30% (12/40) of Flanker scores were at the floor, as were 21% (9/42) of the DCCS scores.”
    Quote in the paperSubstantial claim
    • Rests on an assumption: The NIHTB-CB tasks measure the same cognitive constructs across their different age versions despite differences in stimuli, instructions, and prompts.
  • May combine two claims.

    Findingown data

    There was a positive correlation between age-corrected standard scores on the Flanker and DCCS in individuals with Williams syndrome (rs = 0.58, p < 0.01), but no correlation with age-corrected standard scores on the PSMT (rs = 0.07, p = 0.68).

    “Additionally, there was a positive correlation between age-corrected SS on the Flanker and DCCS (rs= 0.58, p < 0.01), but none with the age-corrected SS on the PSMT (rs = 0.07, p = 0.68).”
    Quote in the paperSubstantial claim
  • Probably not a substantial claim: may not pass S3 (load-bearing).

    Findingnone given

    The current sample comprised a wide age range.

    “In addition, the current sample comprised a wide age range.”
    Quote in the paperNot a substantial claim
  • Probably not a substantial claim: may not pass S3 (load-bearing). May combine two claims.

    Premisecitation

    The Dimensional Change Card Sort Test and the Flanker Inhibitory Control and Attention Test measure aspects of executive functioning, specifically cognitive flexibility (shifting) and inhibitory control/attention (inhibiting), respectively.

    “The Dimensional Card Change Sort Test and the Flanker Inhibitory Control and Attention Test measure aspects of EF, specifically cognitive flexibility (shifting) and inhibitory control/attention (inhibiting) respectively.”
    Quote in the paperNot a substantial claim0 of 1 sources checked
    • cites Zelazo et al., 2013 source
  • Probably not a substantial claim: may not pass S3 (load-bearing). May combine two claims.

    Premisecitation

    The Picture Sequence Memory Test measures episodic memory, an aspect of long-term memory that evidence thus far indicates may be relatively preserved in intellectual disability.

    “The Picture Sequence Memory Test measures episodic memory (Gershon et al., 2013), an aspect of long-term memory that evidence thus far indicates may be relatively preserved in ID (Lifshitz et al., 2011).”
    Quote in the paperNot a substantial claim0 of 2 sources checked
    • cites Gershon et al., 2013 source
    • cites Lifshitz et al., 2011 source
  • Classified as probably not a substantial claim.

    Study design and conductnone given

    Participants who were unable to complete a task for reasons unrelated to their compliance (interruptions in testing, technical difficulties) were removed from the attempted administration total before calculating the proportion of valid administrations.

    “Participants who were unable to complete the task for reasons unrelated to their compliance (i.e., interruptions in testing, technical difficulties) were removed from the attempted administration total before calculating the proportion of valid administrations of each task.”
    Quote in the paperSubstantial claim
  • Probably not a substantial claim: may not pass S3 (load-bearing).

    Interpretationreasoning

    It is possible that scores on the tasks could be slightly elevated as participants who were unable to receive scores may have attained low scores.

    “For this reason, it is possible that scores on the tasks could be slightly elevated as participants who were unable to receive scores may have attained low scores”
    Quote in the paperNot a substantial claim
  • Probably not a substantial claim: may not pass S3 (load-bearing).

    Study design and conductnone given

    The current study did not include information on a variety of other factors that may be related to differences in completion rates or performance, including co-occurring medical problems, psychiatric diagnoses, and medications.

    “Finally, the current study did not include information on a variety of other factors that may be related to differences in completion rates or performance, including co-occurring medical problems, psychiatric diagnoses, and medications.”
    Quote in the paperNot a substantial claim
  • Probably not a substantial claim: may not pass S3 (load-bearing). May combine two claims.

    Premisecitation

    A recent study using the NIHTB-CB in individuals with autism spectrum disorder found administration was feasible, though test completion rates were lower in those with below average IQ compared to average (83% vs. 96% for Flanker, 61% vs. 94% for DCCS, and 48% vs. 69% for PSMT).

    “Additionally, a recent study using the NIHTB-CB in individuals with autism spectrum disorder found administration was feasible, though test completion rates were lower in those with below average IQ compared to average (83% vs. 96% for Flanker, 61% vs. 94% for DCCS, and 48% vs. 69% for Picture Sequence Memory Test [PSMT]; Jones et al., 2021).”
    Quote in the paperNot a substantial claim0 of 1 sources checked
    • cites Jones et al., 2021

      Couldn’t be matched to a paper in Crossref, so it can’t be checked.

  • May combine two claims.

    Interpretationown data

    The rates of incomplete tests were comparable to those in the initial validation studies, indicating the issue is not necessarily specific to WS or ID.

    “however, the rates of incomplete tests were comparable to those in the initial validation studies, indicating the issue is not necessarily specific to WS or ID.”
    Quote in the paperSubstantial claim
  • Probably not a substantial claim: may not pass S3 (load-bearing).

    Study design and conductnone given

    Consent was obtained by participants or caregivers/guardians into an NIH IRB-approved protocol (06-M-0065, NCT00271622).

    “Consent was obtained by participants or caregivers/guardians, depending on status as a minor and cognitive capacity, into an NIH IRB-approved protocol (06-M-0065, NCT00271622).”
    Quote in the paperNot a substantial claim
  • May combine two claims.

    Interpretationreasoning

    The NIHTB-CB uncorrected SS approximates a "person ability score," which indexes an individual's level of functioning relative to the entire normative sample and could potentially be more sensitive to change over time.

    “The NIHTB-CB uncorrected SS approximates a “person ability score,” which indexes an individual’s level of functioning relative to the entire normative sample and could potentially be more sensitive to change over time.”
    Quote in the paperSubstantial claim
  • Probably not a substantial claim: may not pass S3 (load-bearing). May combine two claims.

    Premisecitation

    In individuals with autism spectrum disorder, performance on the NIHTB-CB was associated with full scale IQ and exhibited an effect of diagnosis on certain subscales (Flanker and Pattern Comparison Processing Speed).

    “Performance was associated with full scale IQ and exhibited an effect of diagnosis on certain subscales (Flanker and Pattern Comparison Processing Speed), indicating its potential for uncovering specific cognitive deficits between populations (Jones et al., 2021).”
    Quote in the paperNot a substantial claim0 of 1 sources checked
    • cites Jones et al., 2021

      Couldn’t be matched to a paper in Crossref, so it can’t be checked.

  • Classified as probably not a substantial claim.

    Interpretationreasoning

    It is possible that individuals with ID may not have the reading ability required for the written prompts.

    “This becomes problematic in an ID population, as it is possible that they may not have the reading ability required for the written prompts.”
    Quote in the paperSubstantial claim
  • Classified as probably not a substantial claim.

    Study design and conductown data

    All 47 participants in the present analyses had molecular testing showing, at minimum, the equivalent of FISH positivity for Williams syndrome.

    “All 47 participants in the present analyses have molecular testing showing, at minimum, the equivalent of fluorescent in situ hybridization (FISH) positivity for WS and were enrolled in a study focused on their neurodevelopment.”
    Quote in the paperSubstantial claim
  • Probably not a substantial claim: may not pass S3 (load-bearing).

    Interpretationreasoning

    Administration accommodations such as prompting and redirecting could potentially affect the Flanker and DCCS as their scoring incorporates reaction time into their calculation.

    “although it is noted that administration accommodations such as prompting and redirecting could potentially affect the Flanker and DCCS as their scoring incorporates reaction time into their calculation.”
    Quote in the paperNot a substantial claim
  • May combine two claims.

    Interpretationreasoning

    In individuals with ID, age-corrected standard scores will remain stagnant over time even though performance may improve, unless performance jumps into the range observed in their chronological-aged peers.

    “This results in floor scores, and unless performance jumps into the range observed in their chronological-aged peers, age-corrected standard scores will remain stagnant over time even though performance may improve.”
    Quote in the paperSubstantial claim
    • Rests on an assumption: Age-corrected standard scores reduce the variability in performance of the reference group.
    • Rests on an assumption: Individuals with ID often have performance that is outside the range observed in their chronological-age peers.
  • Classified as probably not a substantial claim.

    Interpretationreasoning

    The better performance on PSMT compared to Flanker and DCCS could be related to the relatively preserved episodic memory seen in WS.

    “which could be related to the relatively preserved episodic memory seen in WS.”
    Quote in the paperSubstantial claim
  • Classified as probably not a substantial claim. May combine two claims.

    Interpretationreasoning

    Administration based on mental age may be helpful in ameliorating the concerns regarding reading level previously mentioned but may be untenable because it requires administering a simultaneous IQ test.

    “Administration based on mental age may be helpful in ameliorating the concerns regarding reading level previously mentioned but may be untenable because it requires administering a simultaneous IQ test.”
    Quote in the paperSubstantial claim
  • Classified as probably not a substantial claim.

    Premisecitation

    As a measure of fluid intelligence, NVIQ is expected to correlate with EF ability, such as the Flanker and DCCS.

    “As a measure of fluid intelligence, it is expected that NVIQ correlates with EF ability, such as the Flanker and DCCS (Blair, 2006; Zelazo et al., 2013).”
    Quote in the paperSubstantial claim0 of 2 sources checked
    • cites Blair, 2006 source
    • cites Zelazo et al., 2013 source
  • May combine two claims.

    Interpretationreasoning

    Comparing performance to a wider age range, such as that used for the uncorrected SS, reduces the frequency of floor scores and allows for within-subject comparison.

    “Comparing that performance to a wider age range, such as that used for the uncorrected SS, reduces the frequency of floor scores, and allows for within-subject comparison.”
    Quote in the paperSubstantial claim
  • Probably not a substantial claim: may not pass S3 (load-bearing), S4 (based).

    Interpretationnone given

    A correlation between uncorrected SS and age is likely.

    “Though a correlation between uncorrected SS and age is also likely”
    Quote in the paperNot a substantial claim
35 of 35
  • The paper relies on this without support.

    The NIHTB-CB tasks measure the same cognitive constructs across their different age versions despite differences in stimuli, instructions, and prompts.

    “These versions are designed to assess the same construct, but use slightly different stimuli, instructions, and prompts throughout the test (e.g., although the 3–7 and 8–11 version of the DCCS contain both an auditory and written prompt for the words “SHAPE” and “COLOR” between trials, the 12+ version only contains the written prompt).”
  • The paper relies on this without support.

    The winsorized floor of the age-corrected standard scores on the NIHTB-CB is one point more extreme than three standard deviations from the mean, an unusually narrow scorable range relative to IQ tests.

    “The winsorized floor of the age-corrected standard scores is one point more extreme than three standard deviations from the mean, an unusually narrow scorable range relative to IQ tests, which usually extend to at least four standard deviations.”
  • The paper relies on this without support.

    For developmental constructs, the variability in performance within a narrow age range is less than that of a sample with a wider age range.

    “For developmental constructs, the variability in performance within a narrow age range will be less than that of a sample with a wider age range.”
  • The paper relies on this without support.

    Age-corrected standard scores reduce the variability in performance of the reference group.

    “Age-corrected standard scores reduce the variability in performance of the reference group”
  • The paper relies on this without support.

    Individuals with ID often have performance that is outside the range observed in their chronological-age peers.

    “individuals with ID often have performance that is outside the range observed in their chronological-age peers.”
  • The paper relies on this without support.

    In individuals with ID, rate of skill development often does not keep up with same-aged peers, resulting in lower scores as they age even though ability level may have remained stable or progressed at a slower rate.

    “This is common in individuals with ID because often their rate of skill development does not keep up with those of their same-aged peers, resulting in lower scores as they age even though their ability level may have remained stable or even progressed at a slower rate than their peers.”
  • The paper relies on this without support.

    Due to ongoing development of EF from early childhood into early adulthood, a positive relationship between EF scores and age is expected when indexing an ability that develops over an extended period.

    “Indeed, due to the ongoing development of EF from early childhood into early adulthood, a positive relationship between EF scores and age is to be expected when indexing an ability that develops over an extended period of the lifespan.”
  • The paper relies on this without support.

    Reaction time is only accounted for in the scoring of the Flanker and DCCS when an accuracy of ≥ 80% is achieved, and thus wouldn’t affect scores for those below this threshold.

    “However, reaction time is only accounted for in the scoring of these tasks when an accuracy of ≥ 80% is achieved, and thus wouldn’t affect scores for those below this threshold.”
  • The paper relies on this without support.

    In the ages 12+ version of the Flanker and DCCS, there are no longer audio prompts that accompany the word prompts that appear on the screen between each trial.

    “However, in the ages 12+ version of the Flanker and DCCS, there are no longer audio prompts that accompany the word prompts that appear on the screen between each trial (“MIDDLE” and “SHAPE”/“COLOR,” respectively).”
  • The paper relies on this without support.

    People with Williams syndrome generally have intellectual disability.

    “people with WS (who generally have ID)”
10 of 10

Statistics recomputed

Every reported test result, recomputed from its test statistic and degrees of freedom. A result shows as not adding up only once an independent check has confirmed it.

  • Consistentχ²(2) = 28.32reported p < .001 · recomputed p < .001

    “χ2(2) = 28.32, p < .001”

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Self-check

Percentages, sums and reported differences re-computed from the paper’s own numbers.

Self-check results appear here once an independent check has confirmed them.

Cites

Flagged first, then by citations

Research works printed but not linked: 2

  1. Kaufman, A., & Kaufman,N. (2004). Kaufman Brief Intelligence Test (KBIT-2). Pearson.
  2. Wechsler, D. (2014). Wisc-V: Technical and Interpretive Manual. Pearson.

Cited by

Still being checked.

Authors
Condy, E. E., Becker, L., Farmer, C., Kaat, A. J., Chlebowski, C., Kozel, B. A., & Thurm, A.
Affiliations
  • National Institute of Mental Health (US)
  • Eunice Kennedy Shriver National Institute of Child Health and Human Development (US)
  • Northwestern University (US)
  • National Heart, Lung, and Blood Institute (US)
Corresponding author
Thurm, Audrey
Venue
American Journal on Intellectual and Developmental Disabilities
Published
2022-10-27
Volume
127
Issue
6
Pages
473-484
ISSN
  • 1944-7558 (ISSN-L)
  • 1944-7515
OpenAlex
W4307475520
Cited by
6
References
39
Citations by year
2026: 1 · 2024: 4 · 2023: 1
FWCI
0.46
Type
journal article
Language
en
Open access
Locations
  • American Journal on Intellectual and Developmental Disabilities (publishedVersion)
  • PubMed (publishedVersion)
  • PubMed Central (acceptedVersion)
Funders
  • National Institutes of Health (ZIA HL 006212)
  • National Heart, Lung, and Blood Institute (ZIAHL006210)
  • National Institute of Mental Health (ZICMH002961)
Topics
  • Life Sciences › Neuroscience › Developmental Neuroscience › Williams Syndrome Research
  • Social Sciences › Psychology › Experimental and Cognitive Psychology › Cognitive Abilities and Testing
  • Social Sciences › Psychology › Neuropsychology and Physiological Psychology › Developmental and Educational Neuropsychology
Keywords
Williams syndrome, NIH Toolbox Cognition Battery, intellectual disability, executive function, flanker task, Dimensional Change Card Sort, episodic memory, floor effect
MeSH terms
Adolescent, Adult, Child, Child, Preschool, Cognition, Feasibility Studies, Humans, Middle Aged, Neuropsychological Tests, Williams Syndrome, Young Adult, Executive Function

About these checks

Registry, retraction, Cites and statistics results are computed, not judged, with one exception: a printed reference that no index lists is read once by a model before it counts as a citation error. Claims are extracted by a model and kept only when their quote appears verbatim in the text; each cited source is then checked, with a model’s help, for whether it says what the claim relies on. Those verdicts can be wrong — read the claim’s quote and the cited source before relying on one.

Claims read by deepseek-v4.1-flash under definition version 3 (extract-v3) on 2026-10-28.

Classified by jev-1.13-20260917 (gold-guideline-v0.2) on 2026-10-28.

Cited sources are checked by gpt-6-sol.

Statistics and arithmetic are recomputed, not judged.

Confidence bands are shown only for fields calibrated on our annotated test set. None is calibrated yet, so no band is shown.

What counts as a claim ›How we measure confidence ›How we check this ›

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