Systematic review and meta-analysis on motor delay prevalence in children with craniosynostosis

UNLABELLED: Craniosynostosis (CS) may influence motor development in childhood through anatomical and functional alterations resulting from premature fusion of cranial sutures. Although several studies have addressed neuropsychomotor outcomes in CS, the lack of consolidated estimates of motor delay prevalence represents a relevant scientific gap, which motivated the present study. This systematic review and meta-analysis aimed to estimate the prevalence of motor development delay in children with CS. Seven electronic databases, gray literature sources, and reference lists of included studies were systematically searched. Observational studies reporting quantitative data on motor performance in children with CS, assessed using standardized instruments, were included. Risk of bias was evaluated using the Joanna Briggs Institute (JBI) Checklist for Prevalence Studies. A total of 8 studies published between 2001 and 2024, involving 510 children with CS, were included. The samples were predominantly non-syndromic, although one study included a small syndromic subgroup (n = 8). The meta-analysis demonstrated a pooled preoperative prevalence of motor delay of 31% (95% CI: 20-44%) with substantial heterogeneity across studies (I = 85.8%). CONCLUSION: Overall, the findings indicate that approximately one in three children with CS presents some degree of motor delay, reinforcing the need for standardized motor assessments, particularly during the preoperative period. WHAT IS KNOWN: • Craniosynostosis (CS) is a congenital condition that may impair neuropsychomotor development due to anatomical and functional brain alterations. • Previous studies have reported heterogeneous findings on motor outcomes in children with CS, but no systematic review or meta-analysis had consolidated the prevalence of motor delay, particularly in the preoperative period. WHAT IS NEW: • This meta-analysis provides the first pooled estimate of motor delay prevalence in children with non‑syndromic CS, showing that approximately one in three children (36%; 95% CI: 28-44%) presents with motor delay before surgery. • The findings highlight the high frequency of motor impairment across all suture subtypes and underscore the urgent need for standardized preoperative motor screening and early multidisciplinary intervention.