Increase of prevalence of craniosynostosis
Open accessAbstract
Introduction Craniosynostosis represents premature closure of cranial sutures. Prevalence is approximately 3.1-6.4 in 10.000 live births, which is reportedly rising. This epidemiologic study aims to provide insight into this rise through an accurate description of the prevalence, exploring regional variation and change over time.Methods The Dutch Association for Cleft Palate and Craniofacial Anomalies was consulted to identify patients with craniosynostosis born between 2008 and 2013. Data were verified using data provided by all hospitals that treated these patients. The following data were collected: date of birth, gender, diagnosis and postal code. Previously reported data from 1997 until 2007 were included to assess for change in prevalence over the years.Results Between 2008 and 2013 759 patients with craniosynostosis were born in the Netherlands. Prevalence of craniosynostosis was 7.2 per 10.000 live births. Sagittal synostosis was the most common form (44%). Poisson regression analysis showed a significant mean annual increase of prevalence of total craniosynostosis (+12.5%), sagittal (+11.7%) and metopic (+20.5%) synostosis from 1997 to 2013.Conclusion The prevalence of craniosynostosis is 7.2 per 10.000 live born children in the Netherlands. Prevalence of total craniosynostosis, sagittal and metopic suture synostosis has risen significantly from 1997 until 2013, without obvious cause.
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The incidence of single suture synostosis increased noticeably and constantly in recent years, with a dynamic change in the frequency of subtypes.
“The incidence of single suture synostosis increased noticeable constant in recent years. Moreover, there is a dynamic change in the frequency of the different subtypes of this clinical picture at the same time [21–24].”
From Developments in diagnostic and surgical techniques in children with sagittal suture craniosynostosis: a systematic review spanning the last 30 years (2025) · cites this paper as [24]
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Self-citationSullivan, L. E., Li, R., Tong, V. S., Jagasia, P. M., Bonfield, C. M., Golinko, M. S., & Pontell, M. E. (2024) · Annals of Plastic Surgery · doi:10.1097/sap.0000000000004131 · Cited by 15 · see citing works
Anderson, M. G., Jungbauer, D., Leclair, N. K., Ahn, E. S., Stoltz, P., Martin, J. E., Hersh, D. S., & Bookland, M. J. (2023) · Neurosurgical FOCUS · doi:10.3171/2023.3.focus2349 · Cited by 14 · see citing works
Chattha, A. S., Bucknor, A. E. M., Curiel, D. A., Ultee, K. H. J., Afshar, S., & Lin, S. J. (2018) · Journal of Craniofacial Surgery · doi:10.1097/scs.0000000000004561 · Cited by 14 · see citing works
The interleukin-11 receptor variant p.W307R results in craniosynostosis in humans
Ahmad, I., Lokau, J., Kespohl, B., Malik, N. A., Baig, S. M., Hartig, R., Behme, D., Schwab, R., Altmüller, J., Jameel, M., et al. (2023) · Scientific Reports · doi:10.1038/s41598-023-39466-y · Cited by 13 · see citing works
Choudhary, A., Edgar, M. C., Raman, S., Alkureishi, L. W. T., & Purnell, C. A. (2023) · The Cleft Palate-Craniofacial Journal · doi:10.1177/10556656231204506 · Cited by 13 · see citing works
The Use of Artificial Intelligence for the Classification of Craniofacial Deformities
Kuehle, R., Ringwald, F. G., Bouffleur, F., Hagen, N., Schaufelberger, M., Nahm, W., Hoffmann, J., Freudlsperger, C., Engel, M., & Eisenmann, U. (2023) · Journal of Clinical Medicine · doi:10.3390/jcm12227082 · Cited by 13 · see citing works
Muenke syndrome: long-term outcome of a syndrome-specific treatment protocol
den Ottelander, B. K., de Goederen, R., van Veelen, M. C., van de Beeten, S. D. C., Lequin, M. H., Dremmen, M. H. G., Loudon, S. E., Telleman, M. A. J., de Gier, H. H. W., Wolvius, E. B., et al. (2019) · Journal of Neurosurgery Pediatrics · doi:10.3171/2019.5.peds1969 · Cited by 13 · see citing works
Self-citationHolle, J. B., Finger, T., Lugonja, J., Schmidt, F., Schaumann, A., Gratopp, A., Thomale, U., von Bernuth, H., & Schulz, M. (2021) · Frontiers in Pediatrics · doi:10.3389/fped.2021.720074 · Cited by 12 · see citing works
Review of Recurrently Mutated Genes in Craniosynostosis Supports Expansion of Diagnostic Gene Panels
Tooze, R. S., Calpena, E., Weber, A. P., Wilson, L. C., Twigg, S. R. F., & Wilkie, A. O. M. (2023) · Genes · doi:10.3390/genes14030615 · Cited by 12 · see citing works
Headache in Postoperative Isolated Sagittal Synostosis
van de Beeten, S. D. C., Mathijssen, I. M. J., Kamst, N. W., & van Veelen, M. C. (2019) · Plastic & Reconstructive Surgery · doi:10.1097/prs.0000000000005481 · Cited by 12 · see citing works
Self-citationCare, H., Kennedy-Williams, P., Cunliffe, A., Denly, S., Horton, J., Kearney, A., Knapp, M., O’Leary, G., Piggott, K., Pinckston, M., et al. (2019) · The Journal of craniofacial surgery (Print) · doi:10.1097/scs.0000000000005575 · Cited by 13 · see citing works
de Planque, C. A., Petr, J., Gaillard, L., Mutsaerts, H. J. M. M., van Veelen, M. C., Versnel, S. L., Dremmen, M. H. G., & Mathijssen, I. M. J. (2022) · Plastic & Reconstructive Surgery · doi:10.1097/prs.0000000000008931 · Cited by 12 · see citing works
Self-citationVasconcellos, F. D. N., Garcia-Torrico, F., Betta, V. H. C., Pedroso, G. M. D., Tomas, B. E. D. A., Vargas, V. P. S., Vilela, M. A. D., Cavalheiro, S., & da Costa, M. D. S. (2025) · Neurosurgery · doi:10.1227/neu.0000000000003502 · Cited by 13 · see citing works
Sagittal craniosynostosis: a utility outcomes study
Kuta, V., McNeely, P. D., Walling, S. A., & Bezuhly, M. (2017) · Journal of Neurosurgery Pediatrics · doi:10.3171/2017.2.peds16567 · Cited by 11 · see citing works
Rachwalski, M., Khonsari, R. H., & D, G. P. M. (2018) · Molecular Syndromology · doi:10.1159/000493535 · Cited by 11 · see citing works
Cho, D., Evans, K. N., Weed, M. C., Lee, A., & Susarla, S. M. (2020) · World Neurosurgery · doi:10.1016/j.wneu.2020.03.079 · Cited by 11 · see citing works
Leclair, N. K., Lambert, W. A., Knopf, J., Stoltz, P., Hersh, D. S., Martin, J. E., & Bookland, M. J. (2021) · Journal of Neurosurgery Pediatrics · doi:10.3171/2021.7.peds21239 · Cited by 12 · see citing works
Gaillard, L., Goverde, A., van den Bosch, Q. C. C., Jehee, F. S., Brosens, E., Veenma, D. C. M., Magielsen, F. J., de Klein, A., Mathijssen, I. M. J., & van Dooren, M. F. (2021) · Frontiers in Pediatrics · doi:10.3389/fped.2021.772800 · Cited by 11 · see citing works
Self-citationLabuschagne, J., Mutyaba, D., Ouma, J., & Dewan, M. C. (2023) · Journal of Neurosurgery: Pediatrics · doi:10.3171/2022.9.peds22227 · Cited by 10 · see citing works
Choi, T. M., Liu, X., Abdel‐Alim, T., van Veelen, M. C., Mathijssen, I. M. J., Wolvius, E. B., & Roshchupkin, G. V. (2023) · Journal of Cranio-Maxillofacial Surgery · doi:10.1016/j.jcms.2023.11.006 · Cited by 10 · see citing works
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Self-citationDoerga, P. N., Rijken, B. F. M., Bredero-Boelhouwer, H., Joosten, K. F. M., Neuteboom, R. F., Tasker, R. C., Dremmen, M. H. G., Lequin, M. H., van Veelen, M. C., & Mathijssen, I. M. J. (2020) · European Journal of Paediatric Neurology · doi:10.1016/j.ejpn.2020.06.018 · Cited by 10 · see citing works
Self-citationVu, G. H., Mazzaferro, D. M., Kalmar, C. L., Zimmerman, C. E., Humphries, L. S., Swanson, J. W., Bartlett, S. P. P., & Taylor, J. A. (2020) · Journal of Craniofacial Surgery · doi:10.1097/scs.0000000000006492 · Cited by 10 · see citing works
Apert syndrome: the Paris and Rotterdam philosophy
Driessen, C., van Veelen, M. C., Joosten, K. F. M., Versnel, S. L., van Nieuwenhoven, C. A., Wolvius, E. B., Bredero-Boelhouwer, H., Arnaud, É., & Mathijssen, I. M. J. (2017) · Expert Opinion on Orphan Drugs · doi:10.1080/21678707.2017.1335195 · Cited by 10 · see citing works
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Agushi, R., Scagnet, M., Spacca, B., Mura, R., Grandoni, M., Mussa, F., & Genitori, L. (2023) · Journal of Neurosurgery Pediatrics · doi:10.3171/2023.2.peds22409 · Cited by 9 · see citing works
Ahammout, C., Perez, F. A., Birgfeld, C. B., Koudstaal, M. J., Ellenbogen, R. G., Lee, A., & Heike, C. L. (2021) · Plastic & Reconstructive Surgery · doi:10.1097/prs.0000000000008056 · Cited by 9 · see citing works
Mechanism of Disease: Recessive ADAMTSL4 Mutations and Craniosynostosis with Ectopia Lentis
Gustafson, J. A., Björk, M., van Ravenswaaij‐Arts, C. M. A., & Cunningham, M. L. (2022) · Case Reports in Genetics · doi:10.1155/2022/3239260 · Cited by 9 · see citing works
Gaillard, L., Tjaberinga, M. C., Dremmen, M. H. G., Mathijssen, I. M. J., & Vrooman, H. A. (2024) · Journal of Anatomy · doi:10.1111/joa.14028 · Cited by 11 · see citing works
Self-citationForehead Widening in Nonsyndromic Scaphocephaly Operated After 12 Months of Age
D, G. P. M., Jing, X. L., Haber, S. E., James, S., Legros, C., Liu, X. X., Khonsari, R. H., Zérah, M., Meyer, P. G., & Arnaud, É. (2020) · Journal of Craniofacial Surgery · doi:10.1097/scs.0000000000006860 · Cited by 9 · see citing works
Prevalence of craniosynostosis in Finland, 1987–2010: A population‐based study
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Utility of Postoperative Fever Workup After Craniosynostosis Repair
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Squamosal Suture Synostosis: An Under-Recognized Phenomenon
Beiriger, J. W., Zhu, X., Bruce, M. K., Irgebay, Z., Smetona, J. T., Losee, J. E., & Goldstein, J. A. (2022) · The Cleft Palate-Craniofacial Journal · doi:10.1177/10556656221100675 · Cited by 8 · see citing works
Radiographic severity is associated with worse executive function in metopic craniosynostosis
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Craniofacial Surgery-Related Hashtag Utilisation on Instagram
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Self-citationEvaluation of ultra‐low‐dose CT with tin filter for craniosynostosis
Tao, W., & Goetti, R. P. (2024) · Journal of Medical Imaging and Radiation Oncology · doi:10.1111/1754-9485.13812 · Cited by 7 · see citing works
Genetic diagnostic yield in an 11-year cohort of craniosynostosis patients
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Self-citation
- Authors
- 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.
- Affiliations
- Erasmus MC (NL)
- Radboud University Medical Center (NL)
- University Medical Center Utrecht (NL)
- Corresponding author
- Cornelissen, Martijn
- Venue
- Journal of Cranio-Maxillofacial Surgery
- Publisher
- Elsevier BV
- Published
- 2016-09
- Volume
- 44
- Issue
- 9
- Pages
- 1273-1279
- ISSN
- 1010-5182 (ISSN-L)
- OpenAlex
- W2467118334
- Semantic Scholar
- 3452666d413921e215d14d3875604fc408621e95
- Cited by
- 226
- References
- 18
- Citations by year
- 2026: 22 · 2025: 21 · 2024: 32 · 2023: 31 · 2022: 37 · 2021: 31 · 2020: 24 · 2019: 11 · 2018: 9 · 2017: 8
- FWCI
- 7.52
- Type
- journal article
- Language
- en
- Open access
- green
- dspace.library.uu.nl
- Licence
- Locations
- Journal of Cranio-Maxillofacial Surgery (publishedVersion)
- PubMed (publishedVersion)
- Research Publications (Maastricht University) (publishedVersion)
- EUR Research Repository (Erasmus University Rotterdam) (publishedVersion)
- Data Archiving and Networked Services (DANS) (submittedVersion)
- EUR Research Repository (Erasmus University Rotterdam) (submittedVersion)
- Utrecht University Repository (Utrecht University) (submittedVersion, other-oa)
- Pure Amsterdam UMC (submittedVersion)
- Radboud Repository (Radboud University) (submittedVersion)
- Research Publications (Maastricht University) (submittedVersion)
- Utrecht University Repository (Utrecht University) (submittedVersion)
- Funders
- Fonds NutsOhra
- Topics
- Life Sciences › Biochemistry, Genetics and Molecular Biology › Genetics › Craniofacial Disorders and Treatments
- Life Sciences › Biochemistry, Genetics and Molecular Biology › Genetics › Cleft Lip and Palate Research
- Health Sciences › Medicine › Surgery › Pectus Deformity Diagnosis and Treatment
- Keywords
- craniosynostosis, prevalence, temporal trends, sagittal synostosis, metopic synostosis, cranial sutures, regional variation, Netherlands
- MeSH terms
- Craniosynostoses, Female, Humans, Infant, Newborn, Male, Netherlands, Prevalence
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