Evidence map›Paper›PMID 40116126›Full record

ArticleAutism research : official journal of the International Society for Autism Research2025

Age-Related Trajectories of Autistic Traits in Children With Angelman Syndrome.

Doesjka A Hagenaar, Sabine E Mous, Leontine W Ten Hoopen, André B Rietman, Kamil R Hiralal, Karen G C B Bindels-de Heus, Pieter F A de Nijs, Theresa C Mohr, Eline J Lens, Manon H J Hillegers and 3 more

Abstract read
In one paragraph

Article in Autism research : official journal of the International Society for Autism Research, 2025. The graph could read no effect estimate from its abstract, so it casts no vote on the map. Cited by 2 papers.

0numbers the graph read from it
0cells of the map it votes in
2citing 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

2 citing papers in PubMed.

  1. Review
  2. 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

13 authors.

Doesjka A HagenaarENCORE Expertise Centre for Neurodevelopmental Disorders, Erasmus MC, Rotterdam, the Netherlands.ORCID 0009-0004-9274-6316
Sabine E MousENCORE Expertise Centre for Neurodevelopmental Disorders, Erasmus MC, Rotterdam, the Netherlands.
Leontine W Ten HoopenENCORE Expertise Centre for Neurodevelopmental Disorders, Erasmus MC, Rotterdam, the Netherlands.ORCID 0000-0002-3122-7713
André B RietmanENCORE Expertise Centre for Neurodevelopmental Disorders, Erasmus MC, Rotterdam, the Netherlands.ORCID 0000-0002-9004-0836
Kamil R HiralalENCORE Expertise Centre for Neurodevelopmental Disorders, Erasmus MC, Rotterdam, the Netherlands.
Karen G C B Bindels-de HeusENCORE Expertise Centre for Neurodevelopmental Disorders, Erasmus MC, Rotterdam, the Netherlands.
Pieter F A de NijsENCORE Expertise Centre for Neurodevelopmental Disorders, Erasmus MC, Rotterdam, the Netherlands.
Theresa C MohrENCORE Expertise Centre for Neurodevelopmental Disorders, Erasmus MC, Rotterdam, the Netherlands.
Eline J LensENCORE Expertise Centre for Neurodevelopmental Disorders, Erasmus MC, Rotterdam, the Netherlands.
Manon H J HillegersDepartment of Child- and Adolescent Psychiatry/Psychology, Erasmus MC, Rotterdam, the Netherlands.
Henriette A MollENCORE Expertise Centre for Neurodevelopmental Disorders, Erasmus MC, Rotterdam, the Netherlands.
Marie-Claire Y de WitENCORE Expertise Centre for Neurodevelopmental Disorders, Erasmus MC, Rotterdam, the Netherlands.
Gwen C DielemanENCORE Expertise Centre for Neurodevelopmental Disorders, Erasmus MC, Rotterdam, the Netherlands.

Funding

Erasmus MC Sophia Foundation
6 · The paper itself

Abstract

Angelman syndrome (AS) is a rare neurogenetic disorder. Previous studies indicate a high prevalence of autism spectrum disorder (ASD) with considerable variability. Little is known regarding the longitudinal trajectory of autistic traits. We aim to investigate autistic traits, the effect of age on these traits, and associated features in AS children. This (partly) longitudinal clinical record study at the ENCORE Expertise Center involved 107 AS children aged 2-18 with one (N = 107), two (N = 49), or three (N = 14) measurements. Autistic traits and sensory processing issues were assessed using various instruments, and DSM classifications were used descriptively. Covariates were genotype, gender, and epilepsy. Results indicate a high prevalence of autistic traits and sensory processing issues. Children with the deletion genotype exhibited more autistic traits. Autism Diagnostic Observation Schedule (ADOS) classifications indicated higher rates of ASD compared to clinician DSM classifications. Autistic traits generally remained stable over time, except that ADOS scores significantly decreased for children with the UBE3A mutation genotype, and in the social affect domain for the entire group. In conclusion, incorporating the assessment of autistic traits and sensory processing into clinical practice for AS is important to inform adaptations of the environment to meet the child's needs. Additionally, clinicians and researchers should be mindful of the potential for overestimating ASD traits in AS when relying on the ADOS. ASD diagnosis in AS should integrate multiple diagnostic instruments, diverse hetero-anamnestic sources, and multidisciplinary expert opinions.

Indexed as

Angelman SyndromeAutism Spectrum DisorderAutistic DisorderAdolescentAge FactorsChildChild, PreschoolFemaleGenotypeHumansLongitudinal StudiesMaleUbiquitin-Protein LigasesUbiquitin-Protein LigasesAngelman syndromeAutism Spectrum Disorderautistic traitslongitudinalrepeated measuressensory processing

Identifiers

PMID40116126
PMCPMC12015797

What OpenQuestion holds

Textmetadata
LicenceCC BY
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.