Risk factors and time trends for isolated craniosynostosis
Open accessAbstract
Background: We sought to investigate associations between maternal/infant characteristics and isolated craniosynostosis as well as its subtypes sagittal, metopic, and coronal synostosis, and assess trends in the prevalence of these conditions. Methods: We identified cases in the Texas Birth Defects Registry from 1999 to 2014. We used Poisson regression to identify associations between maternal/infant characteristics and craniosynostosis. We used joinpoint regression and unadjusted Poisson regression to evaluate temporal trends. Finally, we computed adjusted Poisson models to evaluate whether temporal trends were evident after accounting for changes in the population distributions of maternal/infant characteristics over time. Results: Relative to all live births in the general population, cases were more frequently male or preterm. Mothers of cases were more frequently non‐Hispanic white and more frequently obese. Non‐Hispanic black or Hispanic maternal race/ethnicity was associated with a lower prevalence of all craniosynostosis subtypes. Previous live births were associated with sagittal synostosis; residence on the U.S.‐Mexico border was associated with sagittal and coronal synostosis. The prevalence of any isolated craniosynostosis increased (average annual percent change estimated from joinpoint regression [AAPC]: 2.9%), as did the prevalences of sagittal (AAPC: 3.3%) and metopic synostosis (AAPC: 5.4%). In crude Poisson models, the same temporal trends were observed, however these were attenuated after adjusting for maternal/infant characteristics. Conclusions: Prevalence of isolated craniosynostosis increased from 1999 to 2014. The largest AAPC was observed for metopic synostosis. Changes in the population distribution of associated maternal/infant characteristics may explain these trends.
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Schraw et al. found increased odds of patients coming from parents residing close to the US-Mexico border (aPR=1.29), and Somolan et al. showed a correlation with patients living in more rural areas.
“An example of the link between area of living and CS has been shown by Schraw et al., where they found increased changes of patients to come from parents residing close to the US-Mexico border (aPR = 1.29) [96], and by Somolan et al., showing a correlation with patients living in more rural areas [103].”
From Genetics versus environment in the pathophysiology of sagittal synostosis (2026) · cites this paper as [96]
The Results (Risk Factors for Isolated Craniosynostosis by Subtype) report an aPR of 1.29 for sagittal synostosis among offspring of mothers residing in counties adjoining the U.S.–Mexico border. The citing sentence presents that figure as applying to craniosynostosis generally, but the paper reports a different association for coronal synostosis and none for metopic synostosis. The separately cited claim about rural areas is not attributed to Schraw et al. (LLM, conf=0.88)
Experimental — accuracy not yet measuredLower risks of sCS have been observed in other racial or ethnic groups (OR = 0.6).
“lower risks have been observed in black (OR = 0.2), Hispanic (OR = 0.4), and other racial or ethnic groups (OR = 0.6) [56, 96]”
From Genetics versus environment in the pathophysiology of sagittal synostosis (2026) · cites this paper as [96]
The Results (Risk Factors for Isolated Craniosynostosis; page unavailable) report lower prevalence among offspring of non-Hispanic Black and Hispanic women relative to offspring of non-Hispanic White women. Our copy does not report a lower risk for a separate ‘other’ racial or ethnic group or an OR of 0.6; it describes prevalence ratios rather than odds ratios. (LLM, conf=0.82)
Experimental — accuracy not yet measuredLower risks of sCS have been observed in Hispanic individuals (OR = 0.4).
“lower risks have been observed in black (OR = 0.2), Hispanic (OR = 0.4), and other racial or ethnic groups (OR = 0.6) [56, 96]”
From Genetics versus environment in the pathophysiology of sagittal synostosis (2026) · cites this paper as [96]
The Results section reports lower prevalence of isolated craniosynostosis among offspring of Hispanic women than among offspring of non-Hispanic white women. It describes prevalence ratios, however, and the passages do not substantiate the claimed OR of 0.4. (LLM, conf=0.88)
Experimental — accuracy not yet measuredLower risks of sCS have been observed in black individuals (OR = 0.2).
“lower risks have been observed in black (OR = 0.2), Hispanic (OR = 0.4), and other racial or ethnic groups (OR = 0.6) [56, 96]”
From Genetics versus environment in the pathophysiology of sagittal synostosis (2026) · cites this paper as [96]
The Results section reports lower prevalence of isolated craniosynostosis among offspring of non-Hispanic Black women than among offspring of non-Hispanic White women. It reports prevalence ratios, not the claimed odds ratio of 0.2; the claim also describes the group as Black individuals rather than offspring classified by maternal race/ethnicity. (LLM, conf=0.86)
Experimental — accuracy not yet measuredRace and ethnicity have been reported as modifiers of sagittal craniosynostosis (sCS) risk.
“In addition, race and ethnicity have also been reported as modifiers of sCS risk. Several studies consistently report a higher prevalence among individuals of white race [29, 93], whereas lower risks have been observed in black (OR = 0.2), Hispanic (OR = 0.4), and other racial or ethnic groups (OR = 0.6) [56, 96].”
From Genetics versus environment in the pathophysiology of sagittal synostosis (2026) · cites this paper as [96]
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References 44 · Cited by 22
Research works printed but not linked: 1
- Kapp-Simon KA (1998). Mental development and learning disorders in children with single suture craniosynostosis. Cleft Palate-Craniofacial Journal, 35(3), 197– 203. doi: 10.1597/1545-1569_1998_035_0197_mdaldi_2.3.co_2 9603552 · DOI
- Authors
- Schraw, J. M., Woodhouse, J. P., Langlois, P. H., Canfield, M. A., Scheuerle, A. E., Agopian, A. J., Benjamin, R. H., & Lupo, P. J.
- Affiliations
- Baylor College of Medicine (US)
- Texas Department of State Health Services (US)
- The University of Texas Southwestern Medical Center (US)
- The University of Texas Health Science Center at Houston (US)
- Corresponding author
- Schraw, Jeremy M
- ORCID
- Venue
- Birth Defects Research
- Publisher
- Wiley
- Published
- 2020-10-22
- Volume
- 113
- Issue
- 1
- Pages
- 43-54
- ISSN
- 2472-1727 (ISSN-L)
- PMC
- PMC12912631
- OpenAlex
- W3094513675
- Cited by
- 22
- References
- 44
- Citations by year
- 2026: 2 · 2025: 4 · 2024: 10 · 2023: 1 · 2022: 5
- FWCI
- 0.88
- Type
- journal article
- Language
- en
- Open access
- green
- pmc.ncbi.nlm.nih.gov
- Locations
- Birth Defects Research (publishedVersion)
- PubMed (publishedVersion)
- PubMed Central (publishedVersion)
- Funders
- Eunice Kennedy Shriver National Institute of Child Health and Human Development (5R01HD093660)
- Topics
- Life Sciences › Biochemistry, Genetics and Molecular Biology › Genetics › Craniofacial Disorders and Treatments
- Health Sciences › Medicine › Public Health, Environmental and Occupational Health › Spinal Dysraphism and Malformations
- Life Sciences › Biochemistry, Genetics and Molecular Biology › Genetics › Cleft Lip and Palate Research
- Keywords
- sagittal synostosis, metopic synostosis, coronal synostosis, risk factors, temporal trends, maternal obesity, maternal race/ethnicity, preterm birth, sex differences, birth defects registry, Texas
- MeSH terms
- Craniosynostoses, Ethnicity, Female, Humans, Infant, Infant, Newborn, Male, Mothers, Risk Factors, Prevalence
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