Epidemiology of Hospital Admissions for Craniosynostosis in Australia: A Population-Based Study
Open accessAbstract
To describe trends, age, and sex-specific patterns of population hospital admissions with a diagnosis of craniosynostosis (CS) in Australia. Population data for hospital separations (in-patient) from public and private hospitals (July 1996-June 2018) were obtained from the publicly available Australian Institute of Health and Welfare (AIHW) National Hospital Morbidity Database. The outcome variables were hospital separation rates (HSR) (number of hospital separations divided by the estimated resident population [ERP] per year) and average length of stay (aLOS) (patient days divided by the number of hospital separations) with a diagnosis of CS. Trends in HSR and aLOS adjusted for age, sex, and type of CS were investigated by negative binomial regression presented as annual percent change (APC). In 8057 admissions identified, we observed no significant change in the annual trend for HSR for the 22-year period. However, a marginal annual decrease of 1.6% (95% CI: -0.7, -2.4) in the aLOS was identified for the same time period. HSR were higher for males, infants, and single suture synostosis. aLOS was 3.8 days (95% CI: 3.8, 3.9) per visit, longer for syndromic conditions. There was a minor reduction in the average length of hospital stay for CS over the 22-year period potentially indicative of improved care. Population-level information on hospitalisations for rare craniofacial conditions can inform research, clinical, and surgical practice.
Integrity signals
Registry
Cites
Statistics
Claims the paper makes
Claims other papers make about this one
Each is checked against this paper’s own text: does it say what the citing paper says it does?
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 [74]
Cites
null (2016) · Philosophy Study · doi:10.17265/2159-5313/2016.09.003 · Cited by 28,317 · see citing works
Rath, A., Olry, A., Dhombres, F., Brandt, M., Urbero, B., & Aymé, S. (2012) · Human Mutation · doi:10.1002/humu.22078 · Cited by 367 · see citing works
Mathijssen, I. M. J. (2015) · Journal of Craniofacial Surgery · doi:10.1097/scs.0000000000002016 · Cited by 232 · cited 15× · see citing works
“Furthermore, it will be critical that health system coding (Orphacodes) is granular and can accommodate for clinical grouping that can accommodate meaningful and accurate classification including for syndromic and rare causes of CS.31”
BackgroundMethodInfluentialSagittal craniosynostosis: Surgical outcomes and long-term growth
Fearon, J. A., McLaughlin, E. B., & Kolar, J. C. (2006) · Plastic and Reconstructive Surgery · doi:10.1097/01.prs.0000200774.31311.09 · Cited by 146 · cited 2× · see citing works
“Individuals born with syndromic CS, however, have prolonged service contacts when compared to those born with single suture synostosis.(27,6) As most syndromic CS has an underlying genetic cause, better access to genetic diagnostic testing may provide an opportunity for increasing the precision of service planning.”
BackgroundEsparza, J. M., & Hinojosa, J. (2008) · Child s Nervous System · doi:10.1007/s00381-008-0691-8 · Cited by 145 · cited 1× · see citing works
“syndromic CS could also be influenced by underlying noncraniofacial structural or functional comorbidities.(28,6) We observed a similar length of hospital stay for infants (under 1 year of age) with single suture synostosis in Australia to those undergoing management for non-syndromic CS in the United States of Americ…”
BackgroundChanging Epidemiology of Nonsyndromic Craniosynostosis and Revisiting the Risk Factors
Lee, H. Q., Hutson, J. M., Wray, A. C., Lo, P. A., Chong, D. K., Holmes, A. D., & Greensmith, A. L. (2012) · Journal of Craniofacial Surgery · doi:10.1097/scs.0b013e318252d893 · Cited by 116 · cited 1× · see citing works
“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.”
BackgroundYan, H., Abel, T. J., Alotaibi, N. M., Anderson, M. A., Niazi, T. N., Weil, A. G., Fallah, A., Phillips, J. H., Forrest, C. R., Kulkarni, A. V., et al. (2018) · Journal of Neurosurgery Pediatrics · doi:10.3171/2018.4.peds17729 · Cited by 86 · cited 11× · see citing works
“Furthermore, it will be critical that health system coding (Orphacodes) is granular and can accommodate for clinical grouping that can accommodate meaningful and accurate classification including for syndromic and rare causes of CS.31”
BackgroundInfluentialAge at Time of Craniosynostosis Repair Predicts Increased Complication Rate
Bruce, W. J., Chang, V. A., Joyce, C., Cobb, A. N., Maduekwe, U. I., & Patel, P. A. (2017) · The Cleft Palate-Craniofacial Journal · doi:10.1177/1055665617725215 · Cited by 66 · see citing works
Imaging in craniosynostosis: when and what?
Massimi, L., Bianchi, F., Frassanito, P., Calandrelli, R., Tamburrini, G., & Caldarelli, M. (2019) · Child s Nervous System · doi:10.1007/s00381-019-04278-x · Cited by 66 · cited 1× · see citing works
“These findings are likely a consequence of refined surgical protocols, better anesthetic techniques, improved perioperative management, and surgical planning (to a lesser extent) through the use of enhanced diagnostic techniques (computed tomographic [CT] scans, threedimensional craniofacial analysis).(23,24,22) A lar…”
BackgroundLin, Y., Pan, I.-W. E., Mayer, R. R., & Lam, S. K. (2015) · Neurosurgical FOCUS · doi:10.3171/2015.9.focus15383 · Cited by 64 · cited 1× · see citing works
“We observed a similar length of hospital stay for infants (under 1 year of age) with single suture synostosis in Australia to those undergoing management for non-syndromic CS in the United States of America, reflecting similar treatment practices.(29) These differences in overall HSR could likely be explained by diffe…”
BackgroundAnaesthetic management for craniosynostosis repair in children
Pearson, A., & Matava, C. T. (2016) · BJA Education · doi:10.1093/bjaed/mkw023 · Cited by 42 · cited 1× · see citing works
“Over time, surgical treatment for CS per se, has evolved from strip craniotomies to minimally invasive procedures (endoscopic suture release with helmet modeling) with the latter exhibiting minimal postoperative complications.(5) However, surgical management of CS incurs high costs and lengthy hospital visits which ar…”
MethodEvolving Trends of Gastrostomy Insertion Within a Pediatric Population
Glasson, E. J., Wong, K., Leonard, H., Forbes, D., Ravikumara, M., Mews, C., Jacoby, P. A., Bourke, J., Trollor, J. N., Srasuebkul, P., et al. (2018) · Journal of Pediatric Gastroenterology and Nutrition · doi:10.1097/mpg.0000000000002042 · Cited by 39 · cited 1× · see citing works
“The APC was calculated by exponentiating the beta coefficient of the time variable (ie year group) and subtracting 1.(18) The association of separation rates and aLOS with age (data available: 1996-2018), sex (data available: 1996-2018), and type of CS (data available: 2002-2018) was assessed using the same modeling a…”
MethodSelf-citationGene/environment interactions in craniosynostosis: A brief review
Durham, E. L., Howie, R. N., & Cray, J. (2017) · Orthodontics and Craniofacial Research · doi:10.1111/ocr.12153 · Cited by 36 · cited 1× · see citing works
“Craniosynostosis (CS) (primary) refers to a developmental condition that results from premature fusion of cranial suture/ s prior to completion of brain growth leading to deformation of the cranium and alterations in craniofacial morphology.(1) In Australia, CS occurs in approximately 1 in every 1900 births with the s…”
BackgroundAbraham, P., Brandel, M. G., Ore, C. L. D., Reid, C. M., Kpaduwa, C. S., Lance, S. H., Meltzer, H. S., & Gosman, A. A. (2018) · Annals of Plastic Surgery · doi:10.1097/sap.0000000000001383 · Cited by 30 · cited 1× · see citing works
“These might reflect those admissions related to patterns of diagnosis (delayed diagnosis), increased waiting times between specialist diagnosis and elective surgery, surgical timing of cases (delayed especially for syndromic CS), postsurgical complications, major revision procedures, and/or continued treatment for spe…”
BackgroundDescription of total population hospital admissions for cleft lip and/or palate in Australia
Lo, J. Y. J., Kilpatrick, N. M., Jacoby, P. A., & Slack-Smith, L. M. (2015) · BMC Oral Health · doi:10.1186/s12903-015-0144-1 · Cited by 23 · cited 12× · see citing works
“Furthermore, it will be critical that health system coding (Orphacodes) is granular and can accommodate for clinical grouping that can accommodate meaningful and accurate classification including for syndromic and rare causes of CS.31”
BackgroundMethodInfluentialSelf-citationHospitalisations up to adulthood for children born with orofacial clefts
Bell, J. C., Nassar, N., Turner, R. M., Bower, C. I., Gillett, D. A., McBain, W., & Raynes‐Greenow, C. H. (2015) · Journal of Paediatrics and Child Health · doi:10.1111/jpc.13024 · Cited by 21 · cited 11× · see citing works
“Furthermore, it will be critical that health system coding (Orphacodes) is granular and can accommodate for clinical grouping that can accommodate meaningful and accurate classification including for syndromic and rare causes of CS.31”
MethodInfluentialDavid, D. J., Anderson, P. J., Flapper, W. J., Syme-Grant, J., Santoreneos, S., & Moore, M. H. (2016) · Journal of Craniofacial Surgery · doi:10.1097/scs.0000000000002709 · Cited by 10 · cited 1× · see citing works
“These findings are likely a consequence of refined surgical protocols, better anesthetic techniques, improved perioperative management, and surgical planning (to a lesser extent) through the use of enhanced diagnostic techniques (computed tomographic [CT] scans, threedimensional craniofacial analysis).(23,24,22) A lar…”
Backgroundmanagement of craniosynostosis in Australia and New Zealand
Ziaziaris, W. A., & Marucci, D. D. (2019) · Australasian Journal of Plastic Surgery · doi:10.34239/ajops.v2i1.98 · Cited by 5 · see citing works
Flagged first, then by citations
Cited by
Sorted by citations
Junaid, M. A., Slack-Smith, L. M., Wong, K., Bourke, J., Baynam, G. S., Calache, H., & Leonard, H. (2022) · Pediatric Research · doi:10.1038/s41390-022-02024-9 · Cited by 34 · see citing works
Self-citationHabarth-Morales, T. E., Davis, H. D., Duca, A., Salinero, L. K., Chandragiri, S., Rios-Diaz, A. J., Broach, R. B., Caterson, E. J., & Swanson, J. W. (2024) · Journal of Cranio-Maxillofacial Surgery · doi:10.1016/j.jcms.2024.02.016 · Cited by 4 · 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
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
Psychosocial Support for Australian Families Impacted by Craniosynostosis: A Qualitative Study
Osborn, A. J., Roberts, R., & Dorstyn, D. (2025) · The Cleft Palate-Craniofacial Journal · doi:10.1177/10556656251337824 · Cited by 1 · see citing works
Junaid, M. A., Slack-Smith, L. M., Wong, K., Hewitt, T., Bourke, J., Baynam, G. S., Calache, H., & Leonard, H. (2023) · European Journal of Pediatrics · doi:10.1007/s00431-023-04922-4
“However, we observed an increasing trend in all-cause, CS and nonCS-related hospitalizations in our cohort despite stable trends observed both for reported birth prevalence in WA and national hospital separation data on craniosynostosis [7, 15].”
BackgroundMethodResultInfluentialSelf-citationJunaid, M. A., Slack-Smith, L. M., Wong, K., Hewitt, T., Glasson, E. J., Bourke, J., Baynam, G. S., Calache, H., & Leonard, H. (2023) · The Journal of Pediatrics · doi:10.1016/j.jpeds.2023.113418
Self-citationDhiman, S., Panigrahi, I., Padhi, B. K., Gupta, S., Satapathy, P., Khatib, M. N., Gaidhane, S., Kaur, H., Sharma, M., Zahiruddin, Q. S., et al. (2024) · Research Square · doi:10.21203/rs.3.rs-4443370/v1
Lived experience as guidance: A qualitative study of parental advice for craniosynostosis
Silvestri, T. P., Osborn, A. J., & Roberts, R. (2026) · Journal of Child Health Care · doi:10.1177/13674935261476741
- Authors
- Junaid, M., Slack-Smith, L., Wong, K., Baynam, G., Calache, H., Hewitt, T., & Leonard, H.
- Affiliations
- The Kids Research Institute Australia (AU)
- Deakin University (AU)
- Princess Margaret Hospital for Children (AU)
- Corresponding author
- Junaid, Mohammed
- ORCID
- Venue
- The Cleft Palate Craniofacial Journal
- Publisher
- SAGE Publications
- Published
- 2022-02-07
- Volume
- 60
- Issue
- 5
- Pages
- 569-576
- ISSN
- 1055-6656 (ISSN-L)
- 1545-1569
- OpenAlex
- W4210286432
- Cited by
- 9
- References
- 18
- Citations by year
- 2026: 2 · 2025: 2 · 2024: 2 · 2023: 2 · 2022: 1
- FWCI
- 1.06
- Type
- journal article
- Language
- en
- Open access
- green
- figshare.com
- Locations
- The Cleft Palate-Craniofacial Journal (publishedVersion)
- PubMed (publishedVersion)
- UWA Profiles and Research Repository (UWA) (submittedVersion)
- Figshare (submittedVersion, cc-by)
- Funders
- Stan Perron Charitable Foundation
- Australian Government International RTP Scholarhsip (UWA22647757)
- 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
- Social Sciences › Arts and Humanities › Archeology › Forensic Anthropology and Bioarchaeology Studies
- Keywords
- craniosynostosis, hospitalization, length of hospital stay, temporal trends, sex differences, age differences, syndromic craniosynostosis, craniofacial conditions, Australia
- MeSH terms
- Australia, Craniosynostoses, Female, Hospitalization, Hospitals, Humans, Infant, Length of Stay, Male
About these checks
Registry, retraction, Cites and statistics results are computed, not judged, with one exception: a printed reference that no index lists is read once by a model before it counts as a citation error. Claims are extracted by a model and kept only when their quote appears verbatim in the text; each cited source is then checked, with a model’s help, for whether it says what the claim relies on. Those verdicts can be wrong — read the claim’s quote and the cited source before relying on one.
Statistics and arithmetic are recomputed, not judged.
Confidence bands are shown only for fields calibrated on our annotated test set. None is calibrated yet, so no band is shown.
What counts as a claim ›How we measure confidence ›How we check this ›
Are you an author, or do you see a problem with this page? Request a review