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Midface Hypoplasia and Cranial Base Morphology in Syndromic Craniosynostosis: A Comparative Analysis Study Using a Predictive Regression Model
Published on: November 4, 2025
How does obstructive sleep apnoea evolve in syndromic craniosynostosis? A prospective cohort study
Caroline Driessen1, Koen F M Joosten, Natalja Bannink
1Department of Plastic, Reconstructive, and Hand Surgery, Dutch Craniofacial Centre, Erasmus Medical Centre--Sophia Children's Hospital, Rotterdam, The Netherlands. c.driessen@erasmusmc.nl
Archives of Disease in Childhood
|May 25, 2013
Summary
Obstructive sleep apnoea syndrome (OSAS) is common in children with syndromic craniosynostosis, often improving naturally in the first three years. Early diagnosis is key, as severe OSAS is unlikely to develop later if not present initially.
Area of Science:
- Pediatric Pulmonology
- Craniofacial Surgery
- Sleep Medicine
Background:
- Syndromic craniosynostosis presents unique challenges in pediatric care.
- Obstructive sleep apnoea syndrome (OSAS) is a significant concern in this population.
- Understanding the natural course of OSAS is crucial for timely intervention.
Purpose of the Study:
- To investigate the prevalence and natural history of OSAS in children with syndromic craniosynostosis.
- To identify factors influencing OSAS progression in this cohort.
- To inform clinical management strategies for OSAS in pediatric craniofacial anomalies.
Main Methods:
- Prospective cohort study involving 97 children with syndromic craniosynostosis.
- Level III sleep studies were conducted to assess OSAS severity.
- Longitudinal data analysis using mixed-effects models for untreated patients.
Main Results:
- OSAS prevalence was high at 68% in the studied population.
- The obstructive apnoea-hypopnoea index naturally decreased over time, particularly in the first three years.
- Midface hypoplasia was associated with higher OSAS values and slower improvement.
- Severe OSAS did not develop later in childhood if not present initially.
Conclusions:
- OSAS is highly prevalent in syndromic craniosynostosis and shows some natural improvement.
- Children with Apert or Crouzon/Pfeiffer syndromes experienced less improvement.
- Continuous clinical surveillance is essential for managing OSAS and associated risk factors in these children.
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