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Incidence of Airway Abnormalities in Children With Craniosynostosis
Dana Eitan1, Aditi Bhuskute2, Patrick Scheffler2
1Creighton University School of Medicine.
Insights
Children with syndromic craniosynostosis have significantly higher rates of airway abnormalities, including choanal atresia and tracheostomy. Routine airway evaluation is recommended for these patients.
Area of Science:
- Pediatric Surgery
- Genetics
- Pulmonology
Background:
- Craniosynostosis, a condition causing premature skull fusion, often presents with airway abnormalities in children.
- Previous studies suggest a link between syndromic craniosynostosis and airway issues, but large-scale data were lacking.
Purpose of the Study:
- To investigate the association between syndromic craniosynostosis and airway anomalies using a national inpatient database.
- To compare the incidence of specific airway diagnoses and tracheostomy in syndromic versus nonsyndromic craniosynostosis patients.
Main Methods:
- Retrospective cohort study utilizing the 2016 Healthcare Cost and Utilization Project Kid's Inpatient Database.
- Analysis of demographic data, airway diagnoses, and comorbidities in 4,914 children with craniosynostosis.
Main Results:
- Syndromic craniosynostosis patients (136 children) showed significantly higher rates of choanal atresia (31% vs. 2.5%).
- Syndromic patients were more likely to have laryngomalacia, tracheal stenosis, and tracheomalacia.
- The incidence of tracheostomy was 5.84 times higher in syndromic craniosynostosis patients (28.4% vs. 4.6%).
Conclusions:
- Syndromic craniosynostosis is strongly associated with a higher incidence of choanal atresia and other airway anomalies.
- Routine airway evaluation is warranted for all syndromic craniosynostosis patients.
- Vigilant airway assessment is also advised for nonsyndromic craniosynostosis patients presenting with aerodigestive symptoms.
Introduction:
Children with syndromic craniosynostosis are known to have a high propensity for associated airway abnormalities. However, this has not been investigated using a large-scale national database.
Methods:
For this retrospective cohort study, the 2016 Healthcare Cost and Utilization Project Kid's Inpatient Database was queried for craniosynostosis patients. Data on demographics, airway diagnoses, and comorbidities were analyzed.
Results:
Four thousand nine hundred fourteen children with craniosynostosis with a mean age of 1.7±3.6 years were identified. Of these, 51% were female and 136 children had an associated syndrome. Choanal atresia was present in 31% of patients with an associated syndrome versus 2.5% without. Syndromic patients are 4.59 times more likely (95% CI 2.65-7.94) to have airway anomalies than nonsyndromic patients. After age and sex adjustment, craniosynostosis patients have higher likelihoods of presenting with other anomalies, with syndromic having higher incidences: 5.23 times (95% CI 2.63-10.39) more likely to have laryngomalacia, 18.30 times (95% CI 3.27-102.36) more likely to have tracheal stenosis, and 4.58 times (95% CI 1.36- 15.43) more likely to have tracheomalacia. Incidence of tracheostomy was 5.84 times (95% CI 3.77-9.04) higher in syndromic patients with craniosynostosis. Tracheostomy rates were 28.4% and 4.6% in craniosynostosis patients with and without associated syndrome, respectively.
Conclusion:
Syndromic craniosynostosis patients had significantly higher incidences of choanal atresia and other airway anomalies. Given a high incidence of airway anomalies, syndromic craniosynostosis patients likely warrant routine airway evaluation. Providers should also be vigilant about airway evaluation in patients with nonsyndromic craniosynostosis when aerodigestive symptoms arise.
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