The DOI matches its Crossref record; 9 of the 9 references it cites were found in a registry or an index.
Changing Epidemiology of Nonsyndromic Craniosynostosis and Revisiting the Risk Factors
Abstract
Recent studies in Europe and the United States report increased incidence of metopic synostosis. Whether a similar trend had occurred in Australia remains unknown. This research aimed to determine changes in incidence and subtypes of craniosynostosis in Victoria and to identify perinatal risk factors. A retrospective audit of patients (n = 522) presenting to the Royal Children's Hospital in Melbourne with nonsyndromic craniosynostosis from 1982 to 2008 was undertaken. Perinatal data were sourced from the Victorian Perinatal Data Collection. The changes in incidence of craniosynostosis subtypes were calculated based on Poisson regression, and risk factors for craniosynostosis and subtypes were analyzed by univariate logistic regression analysis. The prevalence of nonsyndromic craniosynostosis was 3.1 in 10,000 live births in Victoria. On average, the incidence of nonsyndromic craniosynostosis increased by 2.5% per year among Victorian live births. Over 25 years, metopic synostosis incidence significantly increased by 7.1% per year in the population of Victoria, outpacing other subtypes. The risk factors for metopic synostosis include being male, multiple births (ie, twins), preterm gestation, low birth weight, high maternal age, and emergency cesarean birth. This study revealed a true increase in incidence of metopic synostosis in Victoria, which could be a result of increased frequency of multiple births, preterm gestation, low birth weight, and high maternal age in the Victorian population from 1982 to 2008. The incidence of other nonsyndromic craniosynostoses, which include sagittal, unicoronal, and multisutural craniosynostoses, however, has remained unchanged.
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“At birth, there are some phenotypes that has been seen to correlate with sCS, from high weight at birth ≥ 4000 g (OR 1.61–2.91) [2, 82] to very low birth weight < 2500 g (OR = 1.5–2.32) [29, 102]), also to breech presentation (OR = 2.39) [63, 102].”
From Genetics versus environment in the pathophysiology of sagittal synostosis (2026) · cites this paper as [82]
In the citing paper
“Firstly, advanced maternal age (> 35 years) has been found to highly correlate with sagittal suture fusion, with an OR ranging from 2.01 to 2.32 [2, 82, 93, 102], while young maternal age has a protective effect OR 0.6 [56].”
From Genetics versus environment in the pathophysiology of sagittal synostosis (2026) · cites this paper as [82]
In the citing paper
“The majority of the studies studied the odds ratio of male occurring CS with an OR ranging from 1.46 to 4.5 (with geometric mean of ~ 2.7) [2, 3, 15, 26, 50, 53, 74, 80, 82, 86, 96–98, 102, 103, 117].”
From Genetics versus environment in the pathophysiology of sagittal synostosis (2026) · cites this paper as [82]
Citations
Cites
A population‐based study of craniosynostosis in metropolitan Atlanta, 1989–2003
Boulet, S. L., Rasmussen, S. A., & Honein, M. A. (2008) · American Journal of Medical Genetics Part A · doi:10.1002/ajmg.a.32208 · Cited by 370
Evolution in the frequency of nonsyndromic craniosynostosis: Clinical article
Di Rocco, F., Arnaud, E., & Renier, D. (2009) · Journal of Neurosurgery: Pediatrics · doi:10.3171/2009.3.peds08355 · Cited by 253
An Epidemiological Study of Nonsyndromal Craniosynostoses
Kolar, J. C. (2010) · Journal of Craniofacial Surgery · doi:10.1097/scs.0b013e3181f6c2fb · Cited by 186
Update on prevention of folic acid‐preventable spina bifida and anencephaly
Bell, K. N., & Oakley, G. P. (2008) · Birth Defects Research Part A Clinical and Molecular Teratology · doi:10.1002/bdra.20504 · Cited by 88
The incidence of craniosynostosis in the Netherlands, 1997–2007
Kweldam, C. F., van der Vlugt, J. J., & van der Meulen, J. J. N. M. (2011) · Journal of Plastic, Reconstructive & Aesthetic Surgery · doi:10.1016/j.bjps.2010.08.026 · Cited by 77
Maternal Drug Use, Fertility Problems, and Infant Craniostenosis
Källén, B. A. J., & Robert‐Gnansia, E. (2005) · The Cleft Palate-Craniofacial Journal · doi:10.1597/04-031.1 · Cited by 46
Williams, C. L., Sutcliffe, A. G., & Sebire, N. J. (2010) · Ultrasound in Obstetrics and Gynecology · doi:10.1002/uog.7589 · Cited by 16
Taylor, L. K., & Tooth, I. (1993) · New South Wales Public Health Bulletin · doi:10.1071/nb93055 · Cited by 6
Cited by
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Increase of prevalence of craniosynostosis
Cornelissen, M., Ottelander, B. D., Rizopoulos, D., van der Hulst, R., Mink van der Molen, A., van der Horst, C., Delye, H., van Veelen, M.-L., Bonsel, G., & Mathijssen, I. (2016) · Journal of Cranio-Maxillofacial Surgery · doi:10.1016/j.jcms.2016.07.007 · Cited by 226
“…single-suture craniosynostosis form is sagittal suture synostosis, comprising approximately 40 e 60% of single-suture craniosynostosis patients (Boulet et al., 2008, Kweldam et al., 2011, Singer et al., 1999, Wilkie et al., 2010, Lee et al., 2012, Selber et al., 2008, van der Meulen et al., 2009).”
van der Meulen, J. (2012) · Child s Nervous System · doi:10.1007/s00381-012-1803-z · Cited by 169
Han, R. H., Nguyen, D. C., Bruck, B. S., Skolnick, G. B., Yarbrough, C. K., Naidoo, S. D., Patel, K. B., Kane, A. A., Woo, A. S., & Smyth, M. D. (2016) · Journal of Neurosurgery: Pediatrics · doi:10.3171/2015.7.peds15187 · Cited by 123
Understanding craniosynostosis as a growth disorder
Flaherty, K., Singh, N., & Richtsmeier, J. T. (2016) · Wiley Interdisciplinary Reviews Developmental Biology · doi:10.1002/wdev.227 · Cited by 131
Global Epidemiology of Craniosynostosis: A Systematic Review and Meta-Analysis
Shlobin, N. A., Baticulon, R. E., Ortega, C. A., Du, L., Bonfield, C. M., Wray, A. C., Forrest, C. R., & Dewan, M. C. (2022) · World Neurosurgery · doi:10.1016/j.wneu.2022.05.093 · Cited by 113
Self-citation
Metadata
- Authors
- Lee, H. Q., Hutson, J. M., Wray, A. C., Lo, P. A., Chong, D. K., Holmes, A. D., & Greensmith, A. L.
- Affiliations
- Royal Children's Hospital (AU)
- Corresponding author
- Hutson, John M.
- Venue
- Journal of Craniofacial Surgery
- Published
- 2012-09
- Volume
- 23
- Issue
- 5
- Pages
- 1245-1251
- ISSN
- 1049-2275 (ISSN-L)
- OpenAlex
- W2049071779
- Cited by
- 116
- References
- 9
- Citations by year
- 2026: 3 · 2025: 8 · 2024: 10 · 2023: 5 · 2022: 16 · 2021: 12 · 2020: 12 · 2019: 6 · 2018: 8 · 2017: 11 · 2016: 7 · 2015: 7 · 2014: 5 · 2013: 4 · 2012: 2
- FWCI
- 3.46
- Type
- journal article
- Language
- en
- Open access
- closed
- Locations
- Journal of Craniofacial Surgery (publishedVersion)
- PubMed (publishedVersion)
- Topics
- Health Sciences › Medicine › Surgery › Craniofacial Disorders and Treatments
- Health Sciences › Medicine › Surgery › Pectus Deformity Diagnosis and Treatment
- Health Sciences › Dentistry › Oral Surgery › Cleft Lip and Palate Research
- Keywords
- nonsyndromic craniosynostosis, metopic synostosis, incidence trends, risk factors, perinatal risk factors, multiple births, low birth weight, maternal age, sagittal synostosis, unicoronal synostosis, Victoria, Australia
- MeSH terms
- Birth Weight, Craniosynostoses, Female, Humans, Infant, Newborn, Male, Maternal Age, Pregnancy, Pregnancy, Multiple, Retrospective Studies, Risk Factors, Victoria, Incidence, Prevalence, Poisson Distribution, Logistic Models, Premature Birth
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