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
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Evaluation of surgical outcomes in craniosynostosis
Maltese, G. (2013) · Gothenburg University Publications Electronic Archive (Gothenburg University)
Lampros, M. G., Αλεξίου, Γ., Sfakianos, G., & Prodromou, N. (2022) · Springer eBooks · doi:10.1007/978-3-030-80522-7_9
Craniosynostosis and Plagiocephaly
Cartwright, C. C., & Chibbaro, P. D. (2017) · Springer eBooks · doi:10.1007/978-3-319-49319-0_3
Unilateral Coronal Craniosynostosis
Massimi, L., & Rocco, C. D. (2020) · Springer eBooks · doi:10.1007/978-3-319-72168-2_59
Oh, C. W. (2013) · Radiopaedia.org · doi:10.53347/rid-24434
Källén, B. A. J. (2013) · Springer eBooks · doi:10.1007/978-3-319-01472-2_26
Wormian bones and nonsyndromic craniosynostosis: A comparative study using computed tomography
Furtado, L. M. F., Filho, J. A. D. C. V., Simas, R. T., Dantas, F., Holliday, J. B., Costa, J. D. S., & dos Santos, A. K. D. (2022) · Archives of Pediatric Neurosurgery · doi:10.46900/apn.v4i2(may-august).108
“For this purpose, some studies have correlated a higher incidence of WB with nonsyndromic CS, which is a disease that progresses with the premature fusion of one or more skull sutures and affects approximately one each of two thousands of live births [11-13].”
BackgroundBackground and Management Principles
Jimenez, D. F. (2022) · Elsevier eBooks · doi:10.1016/b978-0-323-72175-2.00003-1
Concordant non-syndromic sagittal craniosynostoses in dizygotic twins discordant for sex
Assamadi, M., Hadwe, S. E., Barrit, S., Gilis, N., & Witte, O. D. (2022) · Chirurgia · doi:10.23736/s0394-9508.21.05272-4
Craniosynostosis in Isfahan, Iran: A Cross-Sectional Study
Abdali, H., Anaraki, A. G., & Mahdipour, S. (2022) · Journal of Maxillofacial and Oral Surgery · doi:10.1007/s12663-022-01794-3
“However, the metopic type has recently shown an increasing trend in incidence [4] and is getting second place in the order of incidence rates among different types of craniosynostosis [23].”
InfluentialEpidemiologic Assessment of Craniosynostosis in Mississippi’s Pediatric Population from 2015 to 2020
McCandless, M. G., Brown, M. I., Shiflett, J. M., Weaver, K. J., Hoppe, I. C., & Humphries, L. S. (2024) · Southern Medical Journal · doi:10.14423/smj.0000000000001706
McDowell, M. M., Pollack, I. F., Bykowski, M. R., & Goldstein, J. A. (2024) · Springer eBooks · doi:10.1007/978-3-031-69386-1_7
Endoscopic Strip Craniectomy for the Treatment of Single Suture Craniosynostosis
Holste, K. G., Lynes, J. P., & Magge, S. N. (2025) · Advances and technical standards in neurosurgery · doi:10.1007/978-3-031-90762-3_12
Xu, J., Yan, Q., Song, C., Liang, J., Zhao, L., Zhang, X., Weng, Z., Xu, C., Liu, Q., Xu, S., et al. (2021) · Figshare · doi:10.6084/m9.figshare.14795542
Kokitsu‐Nakata, N. M., Virmond, L. D. A., & Serigatto, H. R. (2026) · Springer eBooks · doi:10.1007/978-3-032-12551-4_5
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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