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Midface Hypoplasia and Cranial Base Morphology in Syndromic Craniosynostosis: A Comparative Analysis Study Using a Predictive Regression Model
Published on: November 4, 2025
Neurodevelopmental functioning of infants with untreated single-suture craniosynostosis during early infancy
Annette C Da Costa1, Vicki A Anderson, Ravi Savarirayan
1Department of Plastic and Maxillofacial Surgery, The Royal Children's Hospital, Melbourne, VIC 3052, Australia. annette.dacosta@rch.org.au
Insights
Infants with untreated single-suture craniosynostosis (SSC) show developmental delays, particularly in motor skills. Early intervention is crucial for managing neurodevelopmental impairments in children with SSC.
Area of Science:
- Pediatric neurology
- Craniofacial surgery
- Developmental pediatrics
Background:
- Single-suture craniosynostosis (SSC) is a congenital condition involving premature fusion of one skull suture.
- This fusion restricts cranial and brain growth during a critical developmental period.
- SSC is associated with an increased risk of neurodevelopmental impairment.
Purpose of the Study:
- To investigate the neurodevelopmental outcomes in infants with untreated single-suture craniosynostosis.
- To identify specific areas of developmental delay associated with SSC prior to surgical intervention.
Main Methods:
- A cohort of 56 infants with unoperated SSC (sagittal, metopic, unicoronal) aged 4-16 months was studied.
- Neurodevelopmental functioning was assessed using the Bayley Scales of Infant Development, second edition (Mental Development Index and Psychomotor Development Index).
Main Results:
- Infants with SSC exhibited significantly lower mean mental and motor scores compared to normative data.
- A higher incidence of significant motor developmental delay was observed in the SSC group.
- No cases of accelerated development were noted, and no significant differences were found between SSC subtypes.
Conclusions:
- Untreated single-suture craniosynostosis is linked to a higher prevalence of developmental delay in early infancy.
- Motor skills appear to be particularly vulnerable to impairment in infants with untreated SSC.
- These findings underscore the importance of early assessment and management of SSC for optimal neurodevelopmental outcomes.
Purpose:
Single-suture craniosynostosis (SSC) is a congenital craniofacial disorder, in which premature fusion of one of the skull sutures restricts and distorts growth of the cranium and underlying brain. This disorder of prenatal onset occurs during a critical phase of rapid growth and development of the immature brain. Craniosynostosis carries a known risk of developmental impairment. The neurodevelopmental sequelae of SSC prior to treatment remains however incompletely understood. This study sought to determine the neurodevelopmental sequelae of untreated single-suture craniosynostosis during early infancy.
Methods:
Fifty-six consecutive patients with unoperated SSC (sagittal, metopic and unicoronal) comprised the sample cohort. Patients were aged between 4 and 16 months (M = 8.9 months, SD = 2.9 months). Neurodevelopmental functioning was assessed with the mental (Mental Development Index) and motor (Psychomotor Development Index) scales of the Bayley Scales of Infant Development, second edition.
Results:
Children with SSC displayed significantly lower mean mental (M = 97.7, SD = 6.7, p < 0.05) and motor (M = 87.7, SD = 13.0, p < 0.001) scores than normative population averages. The distribution of these scores also differed significantly from the normative distribution; an increased rate of significant motor developmental delay was found, and none of the children displayed accelerated development. Subgroup comparisons between the primary diagnostic subtypes in this sample revealed no significant differences in mental or motor skill functioning.
Conclusions:
Untreated SSC is associated with an increased incidence of developmental delay during early infancy, with motor skills appearing the most vulnerable to impairment during this developmental phase.
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