Evidence map›Paper›PMID 41820895›Full record

ArticleBMC pediatrics2026

Association between craniosynostosis and neurodevelopmental disorders: a nationwide claims database.

Takanori Yanai, Satomi Yoshida, Koji Kawakami

Abstract read
In one paragraph

Article in BMC pediatrics, 2026. 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

3 authors.

Takanori YanaiDepartment of Pharmacoepidemiology, Kyoto University Graduate School of Medicine and Public Health, Yoshida-Konoecho, Sakyo-ku, Kyoto, 606-8501, Japan.
Satomi YoshidaDivision of Social Medicine, Department of Clinical Medicine, Institute of Medicine, University of Tsukuba, Ibaraki, Japan.
Koji KawakamiDepartment of Pharmacoepidemiology, Kyoto University Graduate School of Medicine and Public Health, Yoshida-Konoecho, Sakyo-ku, Kyoto, 606-8501, Japan. kawakami.koji.4e@kyoto-u.ac.jp.

Funding

Japan Society for the Promotion of Science 25K20597
6 · The paper itself

Abstract

introductionAutism spectrum disorder (ASD) and attention-deficit/hyperactivity disorder (ADHD) in children and have been increasing in recent years, the identification of risk factors and early intervention are important. Cranial deformities may be risk factors for these conditions, but the association with craniosynostosis, especially the non-syndromic form commonly seen in clinical practice, is unclear. Therefore, we examined this association using a nationwide database of 22 million people records.

methodsWe conducted a retrospective cohort study using the JMDC claims database, identifying mother-child pairs born between January 2005 and March 2024 with at least three years of follow-up. Exposure was defined as a diagnosis of non-syndromic craniosynostosis (referred to as craniosynostosis), excluded syndromic such as Apert and Crouzon syndromes. Outcomes were incident ASD and ADHD classified by ICD-10 codes. Cox proportional hazard models adjusted for maternal psychiatric history and neonatal complications calculated hazard ratios (HRs), and a sibling subcohort analysis adjusted for genetic and environmental factors.

resultsAmong 338,117 mother-child pairs, 1,145 children had craniosynostosis. Over a median follow-up of 6.0 years, 16,410 children were diagnosed with ASD and 7,325 ADHD. In adjusted models, craniosynostosis was associated with an increased risk of ASD (HR 2.18, 95% Confidence Interval (CI) 1.80–2.64) and ADHD (HR 2.40, 95%CI 1.77–3.24). Kaplan-Meier curves diverged from early childhood onward, and these findings persisted in sibling analyses.

conclusionCraniosynostosis is significantly associated with the incidence of ASD and ADHD. Physicians and healthcare professionals should recognize cranial anomalies early and conduct careful follow-ups regarding children’s growth.

Indexed as

Attention Deficit Disorder with HyperactivityAutism Spectrum DisorderCraniosynostosesChildChild, PreschoolDatabases, FactualFemaleHumansIncidenceInfantMaleNeurodevelopmental DisordersProportional Hazards ModelsRetrospective StudiesRisk FactorsAttention-deficit/hyperactivity disorder (ADHD)Autism spectrum disorder (ASD)Cohort studyCraniosynostosisNeurodevelopmental disordersRisk factors

Identifiers

PMID41820895
PMCPMC13094190

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Registered trials

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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.