Intellectual and academic functioning of school-age children with single-suture craniosynostosis
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OBJECTIVE: We compared the developmental status of school-age children with single-suture craniosynostosis (case group) and unaffected children (control group). Within the case group we compared the performance of children distinguished by location of suture fusion (sagittal, metopic, unicoronal, lambdoid). METHODS: We administered standardized tests of intelligence, reading, spelling, and math to 182 case participants and 183 control participants. This sample represented 70% of those tested during infancy before case participants had corrective surgery. RESULTS: After adjustment for demographics, case participants' average scores were lower than those of control participants on all measures. The largest observed differences were in Full-Scale IQ and math computation, where case participants' adjusted mean scores were 2.5 to 4 points lower than those of control participants (Ps ranged from .002 to .09). Adjusted mean case-control differences on other measures of achievement were modest, although case deficits became more pronounced after adjustment for participation in developmental interventions. Among case participants, 58% had no discernible learning problem (score <25th percentile on a standardized achievement test). Children with metopic, unicoronal, and lambdoid synostosis tended to score lower on most measures than did children with sagittal fusions (Ps ranged from <.001 to .82). CONCLUSIONS: The developmental delays observed among infants with single-suture craniosynostosis are partially evident at school age, as manifested by lower average scores than those of control participants on measures of IQ and math. However, case participants' average scores were only slightly lower than those of control participants on reading and spelling measures, and the frequency of specific learning problems was comparable. Among case participants, those with unicoronal and lambdoid fusions appear to be the most neurodevelopmentally vulnerable.
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Pathophysiological factors that may explain neurocognitive problems in ISS include increased intracranial pressure, deformation of the brain due to skull deformation, problems with normal brain development, and compression of venous sinuses with impaired venous outflow.
“A number of pathophysiological factors, alone or in combination, may explain the occurrence of neurocognitive problems associated with ISS:increased intracranial pressure [26],deformation of the brain as a result of deformation of the skull [27],problems with the normal development of the brain,compression of the venous sinuses with resulting impairment of venous outflow [14,28].”
From Isolated Sagittal Craniosynostosis: A Comprehensive Review (2024) · cites this paper as [27]
The introductory passage labeled Abstract in our copy says premature suture fusion is believed to constrain or alter brain structures and elevate intracranial pressure, offering possible explanations for neurodevelopmental impairment. It does not discuss compression of venous sinuses or impaired venous outflow, and it does not separately identify disrupted normal brain development as a mechanism. (LLM, conf=0.86)
Experimental — accuracy not yet measuredNeurodevelopment is at least partially impaired in children with ISS, and many authors believe children with ISS are at increased risk of neurodevelopmental disorders, primarily learning difficulties in early childhood and adolescence.
“Nevertheless, neurodevelopment is at least partially impaired in children with ISS, and many authors believe that children with ISS are at increased risk of neurodevelopmental disorders, which manifest primarily in the form of learning difficulties in early childhood and adolescence [9,27].”
From Isolated Sagittal Craniosynostosis: A Comprehensive Review (2024) · cites this paper as [27]
The Conclusions report that developmental delays in children with single-suture craniosynostosis remain partly evident at school age, particularly in IQ and math. The Discussion says several investigators had concluded that many such children have learning disabilities, but this study found only slight evidence of increased learning-problem risk relative to controls. The paper follows children to early elementary school age, not adolescence, and does not establish that difficulties manifest primarily as learning disorders. (LLM, conf=0.86)
Experimental — accuracy not yet measuredIQ scores in children with ISS are usually in the average range, and some children with ISS achieve high or very high IQ scores.
“The scores of the intelligence quotient (IQ) in these children are usually in the average range [27], and there are even studies in which some children with ISS achieved high or very high IQ scores [29,30].”
From Isolated Sagittal Craniosynostosis: A Comprehensive Review (2024) · cites this paper as [27]
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References 48 · Cited by 150
Research works printed but not linked: 1
- [39]Kapp-Simon KA. Mental development and learning disorders in children with single suture craniosynostosis. Cleft Palate Craniofac J. 1998;35(3):197–203 [DOI] [PubMed] [Google Scholar]
- Authors
- Speltz, M. L., Collett, B. R., Wallace, E. R., Starr, J. R., Cradock, M. M., Buono, L., Cunningham, M., & Kapp-Simon, K.
- Affiliations
- Seattle Children's Research Institute (US)
- Harvard University (US)
- St. Louis Children's Hospital (US)
- Children's Healthcare of Atlanta (US)
- Seattle Children's Hospital (US)
- Department of Surgery, Northwestern University, Chicago, Illinois; and
- ORCID
- Venue
- PEDIATRICS
- Publisher
- American Academy of Pediatrics (AAP)
- Published
- 2015-03-01
- Volume
- 135
- Issue
- 3
- Pages
- e615-e623
- ISSN
- 0031-4005 (ISSN-L)
- 1098-4275
- PMC
- PMC4338322
- OpenAlex
- W2168424657
- Cited by
- 150
- References
- 48
- Citations by year
- 2026: 14 · 2025: 17 · 2024: 15 · 2023: 12 · 2022: 17 · 2021: 15 · 2020: 16 · 2019: 11 · 2018: 5 · 2017: 13 · 2016: 10 · 2015: 5
- FWCI
- 7.44
- Type
- journal article
- Language
- en
- Open access
- green
- www.ncbi.nlm.nih.gov
- Locations
- PEDIATRICS (publishedVersion)
- PubMed Central (publishedVersion)
- PubMed (publishedVersion)
- Funders
- National Institutes of Health
- National Institute of Dental and Craniofacial Research (R01DE018227, R01DE013813)
- Topics
- Life Sciences › Biochemistry, Genetics and Molecular Biology › Genetics › Craniofacial Disorders and Treatments
- Health Sciences › Medicine › Surgery › Pectus Deformity Diagnosis and Treatment
- Social Sciences › Arts and Humanities › Archeology › Forensic Anthropology and Bioarchaeology Studies
- Keywords
- single-suture craniosynostosis, elementary school students, intellectual functioning, academic performance, IQ, math computation, reading and spelling, metopic synostosis, unicoronal synostosis, lambdoid synostosis, sagittal synostosis, developmental delay, learning difficulties, neurodevelopmental vulnerability
- MeSH terms
- Achievement, Child, Craniosynostoses, Cross-Sectional Studies, Female, Follow-Up Studies, Humans, Intelligence, Learning, Learning Disabilities, Male, Prognosis, Retrospective Studies, Sutures
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