title: Obstructive sleep apnea-specific quality of life and behavioral problems in children with syndromic craniosynostosis.
abstract: 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.
  
  
  METHODS
  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.
authors.affiliation: Department of Plastic Surgery and Reconstructive Surgery, Erasmus Medical Center-Sophia Children's Hospital, Rotterdam, the Netherlands. n.bannink@erasmusmc.nl
authors.forename: Natalja
authors.initials: N
authors.lastname: Bannink
authors.forename: Marianne
authors.initials: M
authors.lastname: Maliepaard
authors.forename: Hein
authors.initials: H
authors.lastname: Raat
authors.forename: Koen F M
authors.initials: KF
authors.lastname: Joosten
authors.forename: Irene M J
authors.initials: IM
authors.lastname: Mathijssen
mesh_terms: D000293:Adolescent; D002648:Child; D002653:Child Behavior Disorders / Q000209:etiology* / Q000503:physiopathology; D003398:Craniosynostoses / Q000150:complications* / Q000523:psychology; D005260:Female; D006801:Humans; D008297:Male; D017286:Polysomnography; D011446:Prospective Studies; D011788:Quality of Life; D012307:Risk Factors; D012737:Sex Factors; D020181:Sleep Apnea, Obstructive / Q000150:complications* / Q000523:psychology; D013577:Syndrome