Evidence map›Paper›PMID 39407332›Full record

ArticleMolecular autism2024

Developmental trajectories in infants and pre-school children with Neurofibromatosis 1.

Hannah Slevin, Fiona Kehinde, Jannath Begum-Ali, Ceri Ellis, Emma Burkitt-Wright, Jonathan Green, Mark H Johnson, Greg Pasco, Tony Charman, Emily J H Jones and 2 more

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Article in Molecular autism, 2024. The graph could read no effect estimate from its abstract, so it casts no vote on the map. Cited by 6 papers, 1 of them a synthesis that pooled it.

0numbers the graph read from it
0cells of the map it votes in
6citing papers in PubMed, 1 pooled it
–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

6 citing papers in PubMed, 1 synthesis or guideline pooled it.

  1. Pooled it
  2. Review
  3. Article
  4. Review
  5. Article
  6. 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

12 authors.

Hannah Slevin *Division of Psychology and Mental Health, Faculty of Biology, Medicine and Health, University of Manchester, Manchester, UK.
Fiona Kehinde *Division of Psychology and Mental Health, Faculty of Biology, Medicine and Health, University of Manchester, Manchester, UK.
Jannath Begum-AliCentre for Brain and Cognitive Development and Department of Psychological Sciences, Birkbeck, University of London, London, UK.
Ceri EllisDivision of Psychology, Communication and Human Neuroscience, Faculty of Biology, Medicine and Health, University of Manchester, Manchester, UK.
Emma Burkitt-WrightManchester Centre for Genomic Medicine, Manchester University NHS Foundation Trust, Manchester, UK.
Jonathan GreenDivision of Psychology and Mental Health, Faculty of Biology, Medicine and Health, University of Manchester, Manchester, UK.
Mark H JohnsonCentre for Brain and Cognitive Development and Department of Psychological Sciences, Birkbeck, University of London, London, UK.
Greg PascoPsychology Department, Institute of Psychiatry, King's College London, Psychology & Neuroscience, London, UK.
Tony CharmanPsychology Department, Institute of Psychiatry, King's College London, Psychology & Neuroscience, London, UK.
Emily J H JonesCentre for Brain and Cognitive Development and Department of Psychological Sciences, Birkbeck, University of London, London, UK.
Shruti GargDivision of Psychology and Mental Health, Faculty of Biology, Medicine and Health, University of Manchester, Manchester, UK. shruti.garg@manchester.ac.uk.
EDEN-STAARS team

Funding

No grant is acknowledged in the PubMed record.

6 · The paper itself

Abstract

backgroundChildren with Neurofibromatosis 1 (NF1) show cognitive, behavioural and social differences compared to their peers. However, the age and sequence at which these differences begin to emerge is not fully understood. This prospective cohort study examines the cognitive, behavioural, ADHD trait and autism symptom development in infant and pre-school children with NF1 compared with typically developing (TD) children without a family history of neurodevelopmental conditions.

methodsData from standardised tests was gathered at 5, 10, 14, 24 and 36 months of age (NF1 n = 35, TD n = 29). Developmental trajectories of cognitive (Mullen Scales of Early Learning, MSEL) and adaptive behavioural (Vineland Adaptive Behavior Scales, VABS) development from 5 to 36 months were analysed using linear mixed modelling. Measures of ADHD (Child Behavior Checklist) and autism traits (ADOS-2, BOSA-MV and ADI-R) were assessed at 24 and 36 months.

resultsThe developmental trajectory of cognitive skills (all domains of the MSEL) and behavioural skills (four domains of the VABS) differed significantly between NF1 and TD groups. Post-hoc tests demonstrated that the NF1 participants scored significantly lower than TD participants at 24 months on all MSEL and VABS domains. The NF1 cohort demonstrated higher mean autism and ADHD traits at 24 months and 14% of the NF1 cohort met a research diagnostic classification for autism at 36 months. LIMITATIONS: The study has a relatively small sample size due to variable retention and rolling recruitment. Due to limitations imposed by the COVID-19 pandemic, we utilised the Brief Observation of Symptoms of Autism for Minimally Verbal children (BOSA-MV) for some participants, which was administered online and may not gather as accurate a picture of traits as ADOS-2. The BOSA-MV was utilised for 41% of participants with NF1 at 36 months compared to 11% at 24 months. This may explain the reduction in the percentage of children with NF1 that met autism criteria at 36 months.

conclusionsBy 24 months of age, the NF1 cohort show lower cognitive skills and adaptive behaviour and higher levels of autism and ADHD traits as compared to TD children. This has implications for developmental monitoring and referral for early interventions.

trial registrationNot applicable.

Indexed as

CognitionNeurofibromatosis 1Attention Deficit Disorder with HyperactivityAutistic DisorderChild DevelopmentChild, PreschoolFemaleHumansInfantMaleProspective StudiesADHDAutismChildrenCohortNeurofibromatosisNF1Trajectories

Identifiers

PMID39407332
PMCPMC11481376

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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.