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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High weight at birth ≥4000 g is correlated with sCS, with OR 1.61-2.91.
“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]
Advanced maternal age (>35 years) is highly correlated with sagittal suture fusion, with OR ranging from 2.01 to 2.32.
“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]
The majority of studies reported an odds ratio of male occurring CS ranging from 1.46 to 4.5, with a geometric mean of ~2.7.
“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]
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“The surgical intervention involves removing the abnormally fused suture and allowing the cranium to expand in order to accommodate the growing brain.(6) Hence, cranial vault reshaping procedures have shown to have an impact on the neurodevelopmental outcome including speech and language, visuospatial skills, memory, a…”
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Three Patterns of Fronto-Orbital Remodeling for Metopic Synostosis
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“Prematurity, low birth weight, and emergent Caesareans were predictors for all craniosynostosis types.(6) In Philadelphia, a steady rise in the contribution of metopic craniosynostosis patients was demonstrated as well, from 3.”
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“, 2010) followed by metopic, coronal, and lambdoid synostosis according to the last epidemiologic studies (Fonteles, Finnell, George, & Harshbarger, 2016; Lee et al., 2012), respectively .”
Cognitive Development in Single-Suture Craniosynostosis – A Systematic Review
Olsson, K., Engman, J. H., Nowinski, D. J., Ramklint, M., & Frick, M. A. (2023) · Developmental Neuropsychology · doi:10.1080/87565641.2023.2225662 · Cited by 18 · see citing works
“The condition affects approximately one in every 1.300 to 2,500 births (DiRocco et al., 2009; Lee et al., 2012; Tarnow et al., 2022).”
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Les craniosténoses non syndromiques
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“Assessing the relationship between potential risk factors and craniosynostosis, it was found—in accordance with previous studies—that sagittal synostosis was associated with male and twin gestation [Boulet et al., 2008; Lee et al., 2012].”
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Cranioorbital Morphology Caused by Coronal Ring Suture Synostosis
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The Natural History of Cranial Morphology in Sagittal Craniosynostosis
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Role of99mTc-ECD SPECT in the Management of Children with Craniosynostosis
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“About 85% cases are nonsyndromic, occurring as isolated involvement of sutures without any associated extracranial anomaly, whereas remaining 15% occur as syndromic craniosynostosis [4].”
BackgroundGlener, A. D., Allori, A. C., Shammas, R. L., Farjat, A. E., & Marcus, J. R. (2017) · Journal of Craniofacial Surgery · doi:10.1097/scs.0000000000003800 · Cited by 11 · see citing works
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“4 in 1000 births and 80% of cases).(2,3) Syndromic presentations are much less prevalent ranging from 1 in 35 000 (Crouzon syndrome) to 1 in 170 000 (Pfeiffer syndrome) births.”
BackgroundCraniosynostosis: current conceptions and misconceptions
Fonteles, C. S. R., Finnell, R. H., George, T. M., & Harshbarger, R. J. (2016) · Global Medical Genetics · doi:10.3934/genet.2016.1.99 · Cited by 9 · see citing works
“Previous epidemiological work from Australia verified a higher risk of metopic craniosynostosis among males [8]; however, a recent population-based study in the United States failed to show any significant association between metopic synostosis and male gender [7].”
BackgroundResultInfluentialPrevalence of craniosynostosis in Finland, 1987–2010: A population‐based study
Vuola, P., Pakkasjärvi, N. A., Ritvanen, A., Heliövaara, A., Tukiainen, E. J., & Gissler, M. (2024) · Birth Defects Research · doi:10.1002/bdr2.2319 · Cited by 8 · see citing works
“Certain investigations from Europe, Australia, and the United States have documented an increase in the prevalence of craniosynostoses, particularly in relation to meto-pic craniosynostoses (Kweldam et al., 2011; Lee et al., 2012; Selber et al., 2008; van der Meulen et al., 2009).”
BackgroundHead Shape After Sagittal Craniosynostosis Surgery: Open Versus Endoscopic Strip Craniectomy
Crofts, H. J., Mankowski, P., Tamber, M. S., & Courtemanche, D. J. (2023) · Plastic Surgery · doi:10.1177/22925503231184262 · Cited by 8 · see citing works
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“Additionally, there is evidence to suggest that the incidence of metopic synostosis is on the rise (Lee et al., 2012).”
BackgroundInfluentialPlastic and Reconstructive Surgery
Stricker, P. A., Fiadjoe, J. E., & Lerman, J. (2019) · Elsevier eBooks · doi:10.1016/b978-0-323-42974-0.00035-5 · Cited by 8 · see citing works
The Management of Trigonoscaphocephaly as a Result of Combined Metopic and Sagittal Synostosis
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Ganau, M., Syrmos, N. C., & Magdum, S. A. (2022) · Journal of Pediatric Neurosciences · doi:10.4103/jpn.jpn_46_22 · Cited by 7 · see citing works
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Furtado, L. M. F., Filho, J. A. D. C. V., Freitas, L. S., & dos Santos, A. K. D. (2021) · Jornal de Pediatria · doi:10.1016/j.jped.2021.10.004 · Cited by 7 · see citing works
“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…”
ResultPaine, K. M., Tahiri, Y. H., Paliga, J. T., & Taylor, J. A. (2014) · Journal of Craniofacial Surgery · doi:10.1097/scs.0000000000001255 · Cited by 7 · see citing works
“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.”
BackgroundEarly 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 · see citing works
“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.”
BackgroundResultInfluentialCalandrelli, 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 · see citing works
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 · see citing works
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 · see citing works
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 · see citing works
Osborn, A. J., Lange, O., & Roberts, R. (2024) · Developmental Neuropsychology · doi:10.1080/87565641.2024.2357801 · Cited by 6 · see citing works
“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).”
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 · see citing works
“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].”
BackgroundApplication 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 · see citing works
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 · see citing works
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 · see citing works
Yan, Y., McGrath, J. L., Janes, L. E., & Gosain, A. K. (2021) · Journal of Craniofacial Surgery · doi:10.1097/scs.0000000000008061 · Cited by 5 · see citing works
“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…”
BackgroundUnilateral Coronal Craniosynostosis
Massimi, L., & Rocco, C. D. (2017) · Springer eBooks · doi:10.1007/978-3-319-31512-6_59-1 · Cited by 5 · see citing works
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 · see citing works
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 · see citing works
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 · see citing works
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 · see citing works
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 · see citing works
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 · see citing works
Hollamby, A. J., Song, E., Roberts, R., & Osborn, A. J. (2025) · The Journal of craniofacial surgery (Print) · doi:10.1097/scs.0000000000011394
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 · see citing works
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 · see citing works
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 · see citing works
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 · see citing works
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 · see citing works
Magoon, K., Azzolini, A., Yang, R. S., & Taylor, J. A. (2019) · Journal of Craniofacial Surgery · doi:10.1097/scs.0000000000005459 · Cited by 2 · see citing works
Galarza, L. I., Tran, J. N., & Hoppe, I. C. (2022) · Journal of Craniofacial Surgery · doi:10.1097/scs.0000000000008635 · Cited by 2 · see citing works
“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.”
BackgroundEvans, 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 · see citing works
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 · see citing works
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 · see citing works
“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].”
InfluentialFranz‐Odendaal, T. A., & Bezuhly, M. (2021) · Plastic Surgery · doi:10.1177/22925503211057526 · Cited by 1 · see citing works
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 · see citing works
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 · see citing works
Skotting, M. B., Skjøth‐Rasmussen, J., Kofod, T., & Larsen, C. C. (2025) · Ugeskrift for Læger · doi:10.61409/v06240423 · Cited by 1 · see citing works
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 · see citing works
“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 · see citing works
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 · see citing works
Craniosynostosis and Plagiocephaly
Cartwright, C. C., & Chibbaro, P. D. (2012) · Springer eBooks · doi:10.1007/978-3-642-32554-0_3 · Cited by 1 · see citing works
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 · see citing works
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 · see citing works
Shimoji, T. (2017) · Child s Nervous System · doi:10.1007/s00381-017-3526-7 · Cited by 1 · see citing works
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.
- 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
- Life Sciences › Biochemistry, Genetics and Molecular Biology › Genetics › Craniofacial Disorders and Treatments
- Health Sciences › Medicine › Surgery › Pectus Deformity Diagnosis and Treatment
- Life Sciences › Biochemistry, Genetics and Molecular Biology › Genetics › 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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