Cited by 253Craniofacial Disorders and TreatmentsPectus Deformity Diagnosis and TreatmentOrthodontics and Dentofacial Orthopedics

Evolution in the frequency of nonsyndromic craniosynostosis: Clinical article

Di Rocco, Federico, Arnaud, Eric, Renier, Dominique·2009·Journal of Neurosurgery: Pediatrics

Abstract

Object: The prevalence of the different subtypes of craniosynostosis varies greatly. The aim of this study was to analyze the prevalences of the different subtypes of craniosynostosis at a single major craniofacial center and their changes during a 20-year period. Methods: The medical charts of 2808 children hospitalized between 1988 and 2007 for a true craniosynostosis were retrospectively reviewed. Patients were divided according to their subtype of craniosynostosis (sagittal, plagiocephaly, brachycephaly, metopic, oxycephaly, syndromic cases, or unclassifiable). Results: The total number of patients according to their craniosynostosis subtype were as follows: 1224 cases of scaphocephaly, 315 cases of plagiocephaly, 598 cases of trigonocephaly, 105 cases of brachycephaly, 69 cases of oxycephaly, 396 syndromic cases, and 101 unclassifiable cases. The prevalences of these craniosynostoses evolved differently over time. The number of children hospitalized each year increased over time from 106 to 181 cases, but this increase varied according to the subtype of craniosynostosis. Cases of scaphocephaly, plagiocephaly, brachycephaly, and syndromic cases increased similarly (170% increase on average), whereas trigonocephalies dramatically increased (420% increase). Conclusions: The evolution observed in the prevalence of craniosynostosis might result from several factors. An increased recruitment of patients at the center and an improvement in the diagnosis of craniosynostosis might explain the overall increase. However, other mechanisms should be examined to explain the great increase in the number of cases of trigonocephaly (environmental or pharmacological).

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Crossref · 10.3171/2009.3.peds08355

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Research works printed but not linked: 10

  1. [1][1] Ha, John Y., Semerjian, Claire, Tomei, Krystal L., and Wang, Howard D., 2026, "Neurodevelopmental Outcomes Among Nonsyndromic Craniosynostosis Subtypes" Journal of Craniofacial Surgery 1536-3732 Crossref
  2. [2][2] De Gregorio, Valentina, Tosi, Domiziano D., Vita, Alessia, Salvati, Martina, Massimi, Luca, Tamburrini, Gianpiero, Tiberio, Federica, and Lattanzi, Wanda, 2026, "Genetics versus environment in the pathophysiology of sagittal synostosis" Child's Nervous System Vol. 42, No. 1, 1433-0350 Crossref
  3. [3][3] Hong, Zhidan, He, Xuanyi, Duan, Jie, Yu, Fang, Liu, Huanyu, Lu, Dan, Wang, Mei, and Zhang, Yuanzhen, 2024, "Prenatal diagnostic approaches diagnosed craniosynostosis and identified a novel nonsense variant in SMAD6 in a Chinese fetus" Gene Vol. 896, pp 147994, 03781119 Crossref
  4. [5][5] Sakar, Mustafa, Haidar, Hassan, Sönmez, Özcan, Erdoğan, Onur, Saçak, Bülent, Bayri, Yaşar, and Dağçınar, Adnan, 2022, "A new method for quantification of frontal retrusion and complex skull shape in metopic craniosynostosis: a pilot study of a new outcome measure for endoscopic strip craniectomy" Journal of Neurosurgery: Pediatrics Vol. 29, No. 6, pp 650, 1933-0715 Crossref
  5. [6][6] Beuriat, P.A., de Laurentis, C., and Di Rocco, F., 2026, "Anomalies du développement cranial" EMC - Pédiatrie - Maladies infectieuses Vol. 46, No. 3, pp 1, 16375017 Crossref
  6. [7][7] Holste, Katherine, Lynes, John, and Magge, Suresh N., 2025, Advances and Technical Standards in Neurosurgery Vol. 55, pp 209, 978-3-031-90762-3 Crossref
  7. [8][8] Quaresima, Paola, Talamanca, Lorenzo Figà, Dudarewicz, Lech, Russo, Giampiero, Morelli, Michele, Guido, Maurizio, and Agarwal, Umber, 2026, "Supra Occipital Flattening: A Key Sign for Antenatal Diagnosis of Isolated Sagittal Craniosynostosis" Journal of Clinical Ultrasound 1097-0096 Crossref
  8. [9][9] Wei, Yue, Wang, Lei, Liu, Yue, Liu, Kunpeng, and Yao, Lan, 2026, "Perioperative anesthetic considerations and outcomes in Crouzon syndrome: a retrospective analysis of Le Fort III advancement surgery" Frontiers in Pediatrics Vol. 14, 2296-2360 Crossref
  9. [10][10] Aquino, Vincent, Arias, Alondra, Morales, Caleb, Yates, David, and Makoshi, Ziyad, 2026, "Is Endoscopic Treatment of Sagittal Craniosynostosis in Children Aged 6 to 9 Months an Effective Approach for Delayed Presentations?" Journal of Oral and Maxillofacial Surgery 02782391 Crossref
  10. [11][11] Davis, Amani Ali, Zuccoli, Giulio, Haredy, Mostafa M., Runkel, Lauren, Losee, Joseph, Pollack, Ian F., Tamber, Mandeep S., Tyler-Kabara, Elizabeth, Goldstein, Jesse A., and Nischal, Ken-K, 2019, "The Incidence of Chiari Malformations in Patients with Isolated Sagittal Synostosis" Plastic and Reconstructive Surgery - Global Open Vol. 7, No. 2, pp e2090, 2169-7574 Crossref Metrics All Time Past Year Past 30 Days Abstract Views 3615 134 34 Full Text Views 538 6 3 PDF Downloads 512 7 1 EPUB Downloads 0 0 0 PubMed Citation Articles by Federico Di Rocco Articles by Eric Arnaud Articles by Dominique Renier Similar articles in PubMed Article by Federico Di Rocco Article by Eric Arnaud Article by Dominique Renier Similar articles in Google Scholar AANS logo © Copyright 1944-2026 American Association of Neurological Surgeons. All rights reserved, including those for text and data mining, AI training, and similar technologies. Permissions Legal Notices Feedback Editorial Boards CME For Advertisers Job Seekers JNSPG Shop Facebook Twitter Instagram Powered by PubFactory Powered by PubFactory [52.90.36.146] 52.90.36.146 Close My Stuff Sign in Sign up Home COVID-19 Journals Just Published For Authors For Librarians About Us Submit Subscribe Journal of Neurosurgery Journal of Neurosurgery: Spine Journal of Neurosurgery: Pediatrics Journal of Neurosurgery: Case Lessons Neurosurgical Focus Neurosurgical Focus: Video Journal of Neurosurgery Journal of Neurosurgery: Spine Journal of Neurosurgery: Pediatrics Journal Club COVID-19 Leksell Top 25 - Meningioma Leksell Top 25 - Vestibular Schwannoma Leksell Top 25 - Since 2005 Top 25 Cited Gamma Knife® Surgery Articles - Trigeminal Neuralgia Top 25 Cited Gamma Knife® Surgery Articles - Volume 111 Deep Brain Stimulation Best of 2017 Best of 2016 Best of JNSPG - 75th Anniversary Best of 2019 Best of 2020 Parkinson Disease: Top 25 Cited Articles Rhoton Editor's Choice About Us Editorial Boards Publishing Policies JNSPG Staff Job Seekers Contact Us Permissions JNSPG Shop Oxford University Press Partnership ProCite RefWorks Reference Manager BibTeX Zotero EndNote Buy Now

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Authors
Di Rocco, F., Arnaud, E., & Renier, D.
Venue
Journal of Neurosurgery: Pediatrics
Published
2009-07
Volume
4
Issue
1
Pages
21-25
ISSN
  • 1933-0707 (ISSN-L)
  • 1933-0715
OpenAlex
W1599453267
Cited by
253
References
11
Citations by year
2026: 9 · 2025: 13 · 2024: 19 · 2023: 17 · 2022: 18 · 2021: 27 · 2020: 29 · 2019: 21 · 2018: 16 · 2017: 22 · 2016: 16 · 2015: 10 · 2014: 9 · 2013: 6 · 2012: 13
FWCI
3.70
Type
journal article
Language
en
Open access
closed
Locations
  • Journal of Neurosurgery Pediatrics (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
  • Health Sciences › Dentistry › Orthodontics › Orthodontics and Dentofacial Orthopedics
Keywords
nonsyndromic craniosynostosis, prevalence trends, scaphocephaly, trigonocephaly, plagiocephaly, brachycephaly, oxycephaly, syndromic craniosynostosis, pediatric craniofacial surgery, incidence trends
MeSH terms
Child, Craniosynostoses, Humans, Paris, Retrospective Studies, Risk Factors, Prevalence

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