Cited by 116Craniofacial Disorders and TreatmentsPectus Deformity Diagnosis and TreatmentCleft Lip and Palate Research

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Changing Epidemiology of Nonsyndromic Craniosynostosis and Revisiting the Risk Factors

Lee, Hui Qing, Hutson, John M., Wray, Alison C., Lo, Patrick A., Chong, David K., Holmes, Anthony D., Greensmith, Andrew L.·2012·Journal of Craniofacial Surgery

DOI 10.1097/scs.0b013e318252d893

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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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BackgroundInfluential
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Background
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“Background Sagittal synostosis (SS) is the most common form of singlesuture craniosynostosis, with an estimated incidence of 1 in 5000 live births [1,2].”
Background
Maternal occupational exposure to polycyclic aromatic hydrocarbons and craniosynostosis among offspring in the national birth defects prevention study
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“…and all variables were established or suspected risk factors for craniosynostosis (maternal age, maternal education, race/ethnicity, smoking history, exposure to secondhand smoke at home, and body mass index) (Boulet, Rasmussen, & Honein, 2008; Carmichael et al., 2008; Lee et al., 2012).”
Background
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“Presently, metopic synostosis accounts for ~ 30% of single suture craniosynostosis [2,3].”
Background
Multicentre approach to epidemiological aspects of craniosynostosis in Germany
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BackgroundResultInfluential
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Self-citation
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Background
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BackgroundInfluential
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BackgroundResult
CLARITY : Co‐occurrences in achondroplasia—craniosynostosis, seizures, and decreased risk of diabetes mellitus
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“2021;185A:1168–1174.wileyonlinelibrary.com/journal/ajmga Lee et al., 2012).”
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Background
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Background
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BackgroundResultInfluential
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Background
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BackgroundInfluential
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“By contrast, the present study adopted head trauma and CS as the inclusion criteria, with higher incidence rates in males than in females according to the literature, which explains the disparity between the sexes.(18,27) Despite this fact, in a population that consisted mostly of boys, like other studies, our study d…”
Result
Simultaneous Unicoronal and Sagittal Distraction Osteogenesis for the Treatment of Nonsyndromic Multisutural Craniosynostosis
Paine, K. M., Tahiri, Y. H., Paliga, J. T., & Taylor, J. A. (2014) · Journal of Craniofacial Surgery · doi:10.1097/scs.0000000000001255 · Cited by 7
“Multisutural craniosynostosis without an identifiable syndrome or genetic anomaly, also called complex craniosynostosis, is less common and occurs in approximately 8% to 13% of nonsyndromic craniosynostotic cases.(2,3) Bicoronal suture fusion is the most common multisutural craniosynostosis.”
Background
Early Complications and Associated Perioperative Factors in Nonsyndromic Craniosynostosis
Lu, Y., Bao, N., Ghanem, A., Cai, T., Yang, J., & Mu, X. (2017) · Journal of Craniofacial Surgery · doi:10.1097/scs.0000000000003589 · Cited by 7
“The challenge for our future study is to provide data on long-term functional, psychological, and aesthetic outcomes in NSC. ACKNOWLEDGMENT The authors thank Yi Zhang, MS, for structural and grammatical revision of this article.”
BackgroundResultInfluential
Craniofacial Sutural Pattern and Surgical Management in Patients With Different Degrees of Trigonocephaly Severity
Calandrelli, R., Pilato, F., Massimi, L., Panfili, M., Marrazzo, A., Rocco, C. D., & Colosimo, C. (2020) · Journal of Computer Assisted Tomography · doi:10.1097/rct.0000000000001002 · Cited by 7
A Technique for Minimizing the Need for Hemotransfusion in Non-Syndromic Craniosynostosis Surgery
Furtado, L. M. F., Filho, J. A. D. C. V., Hon, Y., Sandes, B. L., Vianna, B. S. B., Valadares, F. W., & dos Santos, A. R. (2020) · Journal of Craniofacial Surgery · doi:10.1097/scs.0000000000006949 · Cited by 7
Incidence of Non-Syndromic and Syndromic Craniosynostosis in Sweden.
Tarnow, P., Kölby, L., Maltese, G., Söfteland, M. B., Lewén, A., Nilsson, P., Enblad, P., & Nowinski, D. (2022) · PubMed · doi:10.1097/scs.0000000000008457 · Cited by 6
A registry study on nonsyndromic craniosynostosis: Long-term associations with academic achievement
Olsson, K., Ramklint, M., Nowinski, D. J., White, R. A., Papadopoulos, F. C., & Frick, M. A. (2024) · Journal of Plastic Reconstructive & Aesthetic Surgery · doi:10.1016/j.bjps.2024.11.014 · Cited by 6
Attention Deficit/Hyperactivity Disorder in Individuals with Non-Syndromic Craniosynostosis: A Systematic Review and Meta-Analysis
Osborn, A. J., Lange, O., & Roberts, R. (2024) · Developmental Neuropsychology · doi:10.1080/87565641.2024.2357801 · Cited by 6
“The prevalence of single suture craniosynostosis is increasing globally, with rates varying between 3 and 8 in every 10,000 live births and a male to female ratio of ~2:1 (Cornelissen et al., 2016; DiRocco et al., 2009; Lee et al., 2012; Tarnow et al., 2022).”
Pre-Operative Ocular Findings and Long-Term Follow-Up in a Large Cohort of Non-Syndromic Unicoronal Craniosynostosis
Rostamzad, P., Esser, Y. S., Tan, E. T. C., Dremmen, M. H. G., Pleumeekers, M. M., & Loudon, S. E. (2023) · Journal of Clinical Medicine · doi:10.3390/jcm12196224 · Cited by 5
“Unicoronal craniosynostosis (UCS), alternatively referred to as anterior plagiocephaly, ranks as the third most prevalent type of non-syndromic craniosynostosis, following sagittal (scaphocephaly) and metopic (trigonocephaly) synostosis [8].”
Background
Application of Computerized Surgical Planning in Craniosynostosis Surgery
Parikh, N., Aral, A. M., Lewis, K., & Alperovich, M. A. (2024) · Seminars in Plastic Surgery · doi:10.1055/s-0044-1786803 · Cited by 5
Longitudinal Three-Dimensional Follow-Up and Secondary Treatment Aspects after Endoscopic and Open Scaphocephaly Surgery
de Jong, G., Meulstee, J. W., van Lindert, E. J., Borstlap, W. A., Maal, T., & Delye, H. (2023) · Plastic & Reconstructive Surgery · doi:10.1097/prs.0000000000010701 · Cited by 5
Advanced parental age: Is it contributing to an increased incidence of non-syndromic craniosynostosis? A review of case-control studies
Abdelhamid, K., Konci, R., ElHawary, H., Gorgy, A., & Smith, L. (2020) · Journal of Oral Biology and Craniofacial Research · doi:10.1016/j.jobcr.2020.11.016 · Cited by 5
Establishment of Objective Clinical Parameters for Assessment of Trigonocephaly: Are Caliper-Derived Clinical Measures Adequate?
Yan, Y., McGrath, J. L., Janes, L. E., & Gosain, A. K. (2021) · Journal of Craniofacial Surgery · doi:10.1097/scs.0000000000008061 · Cited by 5
“However, significantly higher incidences have been reported in recent years with metopic synostosis now accounting for up to 27% of all single suture synostoses, making it the second most common single suture synostosis.(1,2,4,29) This reflects a dramatic increase in incidence from 1 in 15,000 to 1 in 5000, whereas th…”
Background
Unilateral Coronal Craniosynostosis
Massimi, L., & Rocco, C. D. (2017) · Springer eBooks · doi:10.1007/978-3-319-31512-6_59-1 · Cited by 5
An Axin2 mutation and perinatal risk factors contribute to sagittal craniosynostosis: evidence from a Chinese female monochorionic diamniotic twin family
Xu, J., Yan, Q., Song, C., Liang, J., Zhao, L., Zhang, X., Weng, Z., Xu, C., Liu, Q., Xu, S., et al. (2021) · Hereditas · doi:10.1186/s41065-021-00182-0 · Cited by 4
Morphologic Differences between Normocephalic and Scaphocephalic Sagittal Craniosynostosis
Velazquez, A. E., Lebhar, M. S., Yang, J., McCandless, M., Pitre, D. W., Shiflett, M., Weaver, K., Nichols, T., Hoppe, I. C., & Humphries, L. S. (2024) · Plastic and Reconstructive Surgery · doi:10.1097/prs.0000000000011712 · Cited by 2
Prenatal Ultrasound Parameters of Twins With Sagittal Suture Craniosynostosis Question Mechanical Constraint as the Leading Cause
Cinca, K. P., de Planque, C. A., Peters, N. C. J., Versnel, S. L., & Mathijssen, I. M. J. (2022) · Journal of Craniofacial Surgery · doi:10.1097/scs.0000000000008809 · Cited by 3
Epidemiological Characteristics of Children With Non-Cleft Lip/Palate Craniofacial Anomalies
Sawasdipanich, N., Chaithat, B., Rojvachiranonda, N., & Dangsomboon, A. (2021) · Journal of Craniofacial Surgery · doi:10.1097/scs.0000000000008136 · Cited by 3
Associations Between Maternal Periconceptional Alcohol Consumption and Risk of Craniosynostosis Among Offspring, National Birth Defects Prevention Study, 1997–2011
Fisher, S. C., Romitti, P. A., Tracy, M., Howley, M. M., Jabs, E. W., Browne, M. L., & Study, T. N. B. D. P. (2024) · Birth Defects Research · doi:10.1002/bdr2.2420 · Cited by 2
Clinical-Epidemiological Study of a Cohort of 35 Patients with Craniosynostosis
SOMOLAN, C.-V., PETCHESI, C. D., JURCA, C., & BEMBEA, M. (2022) · Maedica - A Journal of Clinical Medicine · doi:10.26574/maedica.2022.17.4.893 · Cited by 2
Diagnostic and Surgical Experiences of Australian Parents Navigating Their Child's Craniosynostosis: A Reflexive Thematic Analysis.
Hollamby, A. J., Song, E., Roberts, R., & Osborn, A. J. (2025) · The Journal of craniofacial surgery (Print) · doi:10.1097/scs.0000000000011394
Can we “overcorrect” our way to normal appearance in metopic craniosynostosis? A single-center’s 33 year odyssey
Hu, A. C., Morales, C. Z., Zhao, A. T., Tolley, P. D., Han, N. A., Ryan, I. A., Romeo, D. J., Swanson, J. W., Bartlett, S. P. P., & Taylor, J. A. (2025) · Journal of Plastic Reconstructive & Aesthetic Surgery · doi:10.1016/j.bjps.2025.11.041 · Cited by 2
Guía de práctica clínica para el diagnóstico, tratamiento y rehabilitación de craneosinostosis no sindrómica en los 3 niveles de atención
Coyotl, D. M. C., Huerta, X. O. R., Vázquez, J. J. S., Sánchez, M. I. D., Peralta, J. S. R., Munive, J. M. T., Porras, M. D. L. P., Cruz, A. J. A., García, J. O. S., & González, C. J. F. (2016) · Cirugía y Cirujanos · doi:10.1016/j.circir.2016.10.028 · Cited by 2
Systematic Review of the Clinical and Experimental Research Assessing the Effects of Craniosynostosis on the Brain
Furtado, L. M. F., Teles, L. R., da Costa, S. A. D. A. M., Matos, V. U. D. S., Teixeira, N. M., Gonçalves, C. A., & Filho, J. A. D. C. V. (2022) · Journal of Craniofacial Surgery · doi:10.1097/scs.0000000000009060 · Cited by 2
Craniofacial Conditions
Fulton, G. K., Blessing, M. S., & Evans, K. N. (2023) · Avery's Diseases of the Newborn · doi:10.1016/b978-0-323-82823-9.00088-x · Cited by 2
Early Bone and Suture Reformations in Different Cranial Regions After Cranial Vault Remodeling for Sagittal Craniosynostosis
Chaisrisawadisuk, S., Phakdeewisetkul, K., Sirichatchai, K., Hammam, E., Prasad, V., & Moore, M. H. (2024) · Journal of Craniofacial Surgery · doi:10.1097/scs.0000000000010508 · Cited by 2
In Situ Hinge Technique for the Frontal Bandeau for the Correction of Metopic Craniosynostosis Reduces Operative Time
Magoon, K., Azzolini, A., Yang, R. S., & Taylor, J. A. (2019) · Journal of Craniofacial Surgery · doi:10.1097/scs.0000000000005459 · Cited by 2
The Simultaneous Use of Spring-Mediated Cranioplasty and Distraction Osteogenesis as a Safe Modality for Multi-Suture Craniosynostosis
Galarza, L. I., Tran, J. N., & Hoppe, I. C. (2022) · Journal of Craniofacial Surgery · doi:10.1097/scs.0000000000008635 · Cited by 2
“The prevalence of multi-sutural fusion is approximately 8% to 13% of nonsyndromic craniosynostotic cases.(4,5) The causes of complex craniosynostoses are not fully understood and are thought to have a multifactorial etiology.”
Background
Craniofacial Malformations
Evans, K. N., Hing, A. V., & Cunningham, M. L. (2018) · Avery's Diseases of the Newborn · doi:10.1016/b978-0-323-40139-5.00100-5 · Cited by 2
Guide to clinical practice for the diagnosis, treatment and rehabilitation of non-syndromic craniosynostosis on 3 levels of care
Coyotl, D. M. C., Huerta, X. O. R., Vázquez, J. J. S., Sánchez, M. I. D., Peralta, J. S. R., Munive, J. M. T., Porras, M. D. L. P., Cruz, A. J. A., García, J. O. S., & González, C. J. F. (2017) · Cirugía y Cirujanos (English Edition) · doi:10.1016/j.circen.2017.11.012 · Cited by 2
Genetics versus environment in the pathophysiology of sagittal synostosis
De Gregorio, V., Tosi, D. D., Vita, A., Salvati, M., Massimi, L., Tamburrini, G., Tiberio, F., & Lattanzi, W. (2026) · Child's Nervous System · doi:10.1007/s00381-026-07396-5 · Cited by 2
“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].”
Influential
Clomiphene Citrate and Enclomiphene Hydrochloride Exposure Is Associated With Interfrontal Suture Fusion in Zebrafish
Franz‐Odendaal, T. A., & Bezuhly, M. (2021) · Plastic Surgery · doi:10.1177/22925503211057526 · Cited by 1
Optimal untwisting of the orbital bandeau in unicoronal craniosynostosis correction: A finite element analysis
Winnand, P., Cevik, E., Ooms, M., Heitzer, M., Bock, A., Hölzle, F., Modabber, A., & Raith, S. (2024) · Journal of the mechanical behavior of biomedical materials/Journal of mechanical behavior of biomedical materials · doi:10.1016/j.jmbbm.2024.106635 · Cited by 1
Morphology and outcomes in combined sagittal and unilateral coronal synostosis
Tandon, D., Burton, J. S., Karsten, M. B., Skolnick, G. B., Naidoo, S. D., Proctor, M. R., Smyth, M. D., & Patel, K. B. (2025) · Journal of Neurosurgery Pediatrics · doi:10.3171/2024.10.peds24245 · Cited by 1
Craniosynostosis
Skotting, M. B., Skjøth‐Rasmussen, J., Kofod, T., & Larsen, C. C. (2025) · Ugeskrift for Læger · doi:10.61409/v06240423 · Cited by 1
Automated Sella-Turcica Annotation and Mesh Alignment of 3D Stereophotographs for Craniosynostosis Patients Using a PCA-FFNN Based Approach
Bielevelt, F., Chargi, N., van Aalst, J. E., Nienhuijs, M. E. L., Maal, T., Delye, H., & de Jong, G. (2025) · Journal of Craniofacial Surgery · doi:10.1097/scs.0000000000011623 · Cited by 1
“4,5 Early diagnosis and intervention are crucial, as they expand therapeutic options and contribute to optimal surgical outcomes.”
Changing concepts in the genetics of craniosynostosis: A pediatric neurology perspective
Fonteles, C. S. R., Khuu, M. B., Bhavani, P., & Finnell, R. H. (2026) · Seminars in Pediatric Neurology · doi:10.1016/j.spen.2026.101330 · Cited by 1
Consistency of Cranial Shape Measures Obtained From Laser Surface and Computed Tomography Imaging
Shock, L. A., Greer, S., Sheahan, L. D., Muzaffar, A. R., & Aldridge, K. J. (2021) · Journal of Craniofacial Surgery · doi:10.1097/scs.0000000000007885 · Cited by 1
Craniosynostosis and Plagiocephaly
Cartwright, C. C., & Chibbaro, P. D. (2012) · Springer eBooks · doi:10.1007/978-3-642-32554-0_3 · Cited by 1
Defining the Normal Range of Forehead Convexity in Infants
Tapp, A., Mantilla‐Rivas, E., Hyman, T. S., García‐Mato, D., Rana, S., Bradford, P. S., Oh, N., Zhang, A., Johnson, K. N., Keating, R., et al. (2025) · Journal of Craniofacial Surgery · doi:10.1097/scs.0000000000011741 · Cited by 1
Surgical Technique in Pediatric Craniofacial Surgery
Furtado, L. M. F., Filho, J. A. D. C. V., dos Santos, A. K. D., Simas, R. T., & Ferreira, J. P. U. (2022) · Archives of Pediatric Neurosurgery · doi:10.46900/apn.v4i2(may-august).111 · Cited by 1
Reply to Dr. Ijichi’s group
Shimoji, T. (2017) · Child s Nervous System · doi:10.1007/s00381-017-3526-7 · Cited by 1
A novel AXIN2 mutation and perinatal risk factors contribute to sagittal craniosynostosis: evidence from a monochorionic diamniotic twin familyPreprint
Xu, J. Y., Yan, Q., Song, C., Liang, J., Zhao, L., Zhang, X., Weng, Z., Xu, C. S., Liu, Q., Xu, S., et al. (2020) · Research Square · doi:10.21203/rs.3.rs-57248/v1
“Non-genetic risk factors, such as intrauterine constraint, twin gestation, low birth weight, malnutrition, premature delivery, maternal thyroid disorders, gestational diabetes, virus infectious, can either cause or exacerbate craniosynostosis[7, 11-14].”
Background

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.
ORCID: Hutson, John M.
Chong, David K.
Venue: Journal of Craniofacial Surgery
Publisher: Ovid Technologies (Wolters Kluwer Health)
Published: 2012-09
Volume: 23
Issue: 5
Pages: 1245-1251
ISSN: 1049-2275 (ISSN-L)
DOI: 10.1097/scs.0b013e318252d893
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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Changing Epidemiology of Nonsyndromic Craniosynostosis and Revisiting the Risk Factors

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