Evidence map›Paper›PMID 41083159›Full record

ArticleJournal of intellectual disability research : JIDR2025

Criterion Validity, Scalability and Stability of Scoring on the Bayley-III in Children With Angelman Syndrome.

Maartje Ten Hooven-Radstaake, Sabine Herrman-Mous, Anjali Sadhwani, Anne Wheeler, Margaret DeRamus, Gwen Dieleman, Cindy Navis, Jeroen Legerstee, Leontine Ten Hoopen, Jan van der Ende and 4 more

Abstract readMulticenter StudyValidation Study
In one paragraph

Article in Journal of intellectual disability research : JIDR, 2025. The graph could read no effect estimate from its abstract, so it casts no vote on the map. Cited by 1 paper.

0numbers the graph read from it
0cells of the map it votes in
1citing papers in PubMed
–field-weighted citation impact
1 · What the graph read from it

What it found

Each row is one number read from the abstract, on the scale the paper reported it, with its interval. Left of the dashed line favours the treatment, right favours the comparator. Under each row is the sentence it came from. New to these charts? A ten-minute tutorial.

The abstract states no effect estimate the extractor could read, or names no intervention and outcome on the map, so this paper lights no cell and moves no belief. It is still indexed, cited and linked below.

2 · The registry

The trial behind it

Trials whose registry record cites this paper, or whose number appears in the abstract. A trial that started after this paper was published is citing it as background, not reporting it.

Neither the registry nor the abstract names a trial number. If this is a trial report, that itself is worth knowing.

3 · Its place in the literature

Who cites it

1 citing paper in PubMed.

  1. Article
4 · The record

Corrections and comments

PubMed lists nothing against this paper. Absence here is not a guarantee, only a check that was made.

5 · Who and what money

Authors and funding

14 authors.

Maartje Ten Hooven-RadstaakeErasmus MC Center of Expertise for Neurodevelopmental Disorders (ENCORE), Rotterdam, the Netherlands.ORCID 0009-0009-0420-7838
Sabine Herrman-MousErasmus MC Center of Expertise for Neurodevelopmental Disorders (ENCORE), Rotterdam, the Netherlands.
Anjali SadhwaniBoston Children's Hospital, Boston, Massachusetts, USA.ORCID 0000-0003-0938-0591
Anne WheelerRTI International, Research Triangle Park, North Carolina, USA.ORCID 0000-0003-2693-5616
Margaret DeRamusCarolina Institute for Developmental Disabilities, University of North Carolina, Chapel Hill, Chapel Hill, North Carolina, USA.
Gwen DielemanErasmus MC Center of Expertise for Neurodevelopmental Disorders (ENCORE), Rotterdam, the Netherlands.
Cindy NavisErasmus MC Center of Expertise for Neurodevelopmental Disorders (ENCORE), Rotterdam, the Netherlands.
Jeroen LegersteeErasmus MC Center of Expertise for Neurodevelopmental Disorders (ENCORE), Rotterdam, the Netherlands.
Leontine Ten HoopenErasmus MC Center of Expertise for Neurodevelopmental Disorders (ENCORE), Rotterdam, the Netherlands.ORCID 0000-0002-3122-7713
Jan van der EndeDepartment of Child and Adolescent Psychiatry/Psychology, Erasmus Medical Center Sophia Children's Hospital, Rotterdam, the Netherlands.
Casey OkoniewskiRTI International, Research Triangle Park, North Carolina, USA.
Laura HirumaCarolina Institute for Developmental Disabilities, University of North Carolina, Chapel Hill, Chapel Hill, North Carolina, USA.
ENCORE group
André RietmanErasmus MC Center of Expertise for Neurodevelopmental Disorders (ENCORE), Rotterdam, the Netherlands.

Funding

Angelman Syndrome and Biomarker Outcome Measure Consortium (ABOM) 2017-002-A
6 · The paper itself

Abstract

backgroundThe Bayley Scales of Infant Development is used in many studies and clinical trials in children with developmental disabilities, including children with Angelman syndrome (AS).

methodWe assessed 142 children with AS in an international multicentre study with the Bayley Scales of Infant Development III, of which 52 children were tested more than once. We assessed criterion validity using proportion analysis, scalability using Mokken analyses and stability of scoring by counting pass-to-fail and fail-to-pass items.

resultsResults revealed good scalability in all scales but the expressive language scale, indicating that the items of these scales measured one underlying trait. In the expressive language scale, the AS-related speech difficulties invalidated scoring. Scoring within children across assessments was unstable for all scales except the gross motor scale, as more than half of the children made one or more errors in previously correct items. Loss or regression of skills does not fully explain this finding. Alternative explanations including motivation, concentration, on-task behaviour and anxiety should also be considered when scores decline.

conclusionsThis study shows that caution should be taken when interpreting single and successive scores of children with AS on the Bayley-III and that other forms of assessment should complement assessment in children with AS.

Indexed as

Angelman SyndromeNeuropsychological TestsChildChild, PreschoolFemaleHumansInfantMalePsychometricsReproducibility of ResultsAngelman syndromeassessmentdevelopmental disabilityecological validity

Identifiers

PMID41083159
PMCPMC12580475

What OpenQuestion holds

Textmetadata
LicenceCC BY-NC-ND
Read underepoch 390

Registered trials

None linked

Read under generation 80e0d062 · epoch 390. Bibliography from PubMed, PubMed Central and OpenAlex; grants from NIH RePORTER; trial links from ClinicalTrials.gov; estimates, votes and beliefs from the OpenQuestion graph.