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Global Epidemiology of Craniosynostosis: A Systematic Review and Meta-Analysis
Nathan A Shlobin1, Ronnie E Baticulon2, Carlos A Ortega3
1Department of Neurological Surgery, Feinberg School of Medicine, Northwestern University, Chicago, Illinois, USA.
Insights
Craniosynostosis, a condition causing craniofacial deformities, affects approximately 5.9 in 10,000 newborns globally. This meta-analysis estimates over 84,000 children worldwide were born with craniosynostosis in 2019.
Area of Science:
- Medical Science
- Epidemiology
- Genetics
Background:
- Craniosynostosis is a congenital condition characterized by the premature fusion of skull sutures, leading to craniofacial deformities.
- Untreated craniosynostosis can cause increased intracranial pressure, neurodevelopmental delays, and psychosocial challenges.
- The global prevalence and incidence of craniosynostosis remain incompletely understood.
Purpose of the Study:
- To conduct a meta-analysis to estimate the global birth prevalence of craniosynostosis.
- To estimate the annual number of children born worldwide with craniosynostosis.
Main Methods:
- A systematic literature search was performed across PubMed, Embase, and Scopus databases.
- Included studies were screened by title, abstract, and full text, with meta-analysis applied to birth prevalence data.
- Global birth prevalence was combined with 2019 birth metrics to estimate worldwide incidence.
Main Results:
- Twenty-four studies were included, providing data on overall and nonsyndromic craniosynostosis.
- The overall birth prevalence was estimated at 5.9 per 10,000 live births (95% CI: 3.9–8.4).
- An estimated 84,665 children were born globally with craniosynostosis in 2019, including 72,857 with nonsyndromic forms.
Conclusions:
- Craniosynostosis is a significant global health concern impacting neurocognitive and craniofacial development.
- Further research and clinical care capacity are needed, especially in low- and middle-income countries.
Objective:
Craniosynostosis leads to craniofacial deformity and may result in raised intracranial pressure, neurocognitive deficits, and psychosocial issues if left untreated. The global epidemiology of craniosynostosis is unknown. We conducted a meta-analysis to estimate global birth prevalence.
Methods:
PubMed, Embase, and Scopus were searched. Articles were screened by title and abstract and then full text. Meta-analysis of birth prevalence was conducted. Birth prevalence figures were combined with metrics detailing the number of births in 2019 to estimate the number of children worldwide born with craniosynostosis annually.
Results:
Of 1378 resultant articles, 24 studies were included, including 20 providing data for craniosynostosis overall and 9 for nonsyndromic craniosynostosis. World Health Organization regions of included studies were 9 (37.5%) European Region, 8 (33.3%) Region of the Americas, 4 (16.7%) Western Pacific region, 2 (8.3%) African Region, and 1 (4.2%) Eastern Mediterranean Region. Lower middle-income countries represented only 4% of study manuscripts. The overall birth prevalence of craniosynostosis was 5.9 per 10,000 live births (20 studies; 95% confidence interval [CI]: 3.9, 8.4; I2 = 100%). The birth prevalence of nonsyndromic craniosynostosis was 5.2 per 10,000 live births (9 studies; 95% CI: 3.4, 7.3; I2 = 98%). The number of children born globally with craniosynostosis in 2019 was estimated to be 84,665 (95% CI: 55,965, 120,540), including 72,857 (95% CI: 47,637, 120,280) with nonsyndromic craniosynostosis.
Conclusions:
Craniosynostosis is a common condition that affects the neurocognitive and craniofacial skeletal development of children worldwide. Initiatives to scale up capacity for craniosynostosis epidemiologic research and clinical care are warranted, particularly in low- and middle-income countries.
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