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Obstructive sleep apnea-specific quality of life and behavioral problems in children with syndromic craniosynostosis
Natalja Bannink1, Marianne Maliepaard, Hein Raat
1Department of Plastic Surgery and Reconstructive Surgery, Erasmus Medical Center-Sophia Children's Hospital, Rotterdam, the Netherlands. n.bannink@erasmusmc.nl
Insights
Children with syndromic craniosynostosis experience lower quality of life due to obstructive sleep apnea (OSA). Behavioral issues are more prevalent in Apert and Muenke syndromes, with OSA severity correlating to these impacts.
Area of Science:
- Pediatric Health
- Sleep Medicine
- Genetics
Background:
- Syndromic craniosynostosis impacts quality of life and may be associated with behavioral problems.
- Obstructive sleep apnea (OSA) is a potential complication in these children.
Purpose of the Study:
- Evaluate the impact of syndromic craniosynostosis on quality of life.
- Assess the prevalence of behavioral problems in syndromic craniosynostosis.
- Determine the impact of OSA in syndromic craniosynostosis compared to controls.
Main Methods:
- Prospective study involving 119 syndromic craniosynostosis patients and 459 controls.
- Utilized Obstructive Sleep Apnea-18 (OSA-18) and Child Behavior Checklist (CBCL) surveys.
- Polysomnography was used to diagnose OSA in the craniosynostosis group.
Main Results:
- Syndromic craniosynostosis patients had higher OSA-18 scores (sleep disturbance, physical suffering, caregiver concerns) than controls.
- Behavioral problems were observed in 67% of boys with Apert and 50% with Muenke syndrome.
- OSA severity correlated significantly with OSA-18 and CBCL scores, with moderate OSA linked to higher sleep disturbance and physical suffering.
Conclusions:
- Obstructive sleep apnea significantly lowers quality of life in children with syndromic craniosynostosis.
- Behavioral problems are more common in boys with Apert and Muenke syndromes.
- OSA-18 and CBCL scores reflect OSA severity in this population.
Objective:
This study aimed at evaluating the impact of syndromic craniosynostosis on quality of life, assessing the association between the presence of craniosynostosis syndrome and prevalence of behavioral problems and assessing the impact of obstructive sleep apnea (OSA) in syndromic craniosynostosis compared with healthy controls.
Method:
A prospective study was carried out using the Obstructive Sleep Apnea-18 (OSA-18) survey and Child Behavior Checklist (CBCL) in 119 syndromic craniosynostosis patients and the OSA-18 survey in 459 controls. The craniosynostosis population underwent a polysomnography to diagnose OSA.
Results:
The total OSA-18 score and scores on the domains sleep disturbance, physical suffering, and caregiver concerns were significantly higher in the craniosynostosis group than in controls. Subgroup analysis revealed behavioral problems in 67% and 50% of boys with Apert and Muenke syndrome, respectively. Correlations between obstructive apnea-hypopnea index and total OSA-18 and CBCL scores were significant. Mean scores for the domains sleep disturbance and physical suffering were significantly higher in moderate OSA.
Conclusions:
OSA is related with a lower quality of life in children with syndromic craniosynostosis. Behavioral problems were more common in boys with Apert and Muenke syndrome. OSA-18 and CBCL scores were correlated with OSA severity.
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