title: Symptoms of sleep disordered breathing in children with craniofacial malformations.
abstract: OBJECTIVE
  The purpose of this study was to investigate the frequency of sleep disordered breathing (SDB) symptoms in a clinical sample of children with congenital craniofacial malformations (CFM) followed at a tertiary medical center and non-selected for sleep problems.
  
  
  METHODS
  Cross-sectional study of 575 children aged 2-18 years followed at the Craniofacial Anomalies Program between March 2007 and May 2011. The Sleep-Related Breathing Disturbance scale of the Pediatric Sleep Questionnaire was used to screen for SDB, snoring, and sleepiness. A cutoff value ≥ 0.33 of the total answered questions identified children with positive screening for SDB symptoms.
  
  
  RESULTS
  Overall, 25% of children screened positive for SDB, 28% for snoring, and 20% for sleepiness. In children with non-syndromic CFM, those with Robin sequence had the highest frequency of SDB, snoring, and sleepiness (43%, 44%, and 38%, respectively). In children with syndromic CFM, velocardiofacial/ DiGeorge syndrome had the highest frequency of SDB and sleepiness (48% and 43%, respectively). Children with Treacher Collins had the highest frequency of snoring (83%). The presence of cleft palate was not associated with an increased frequency of SDB symptoms. Nevertheless, children with syndromic CFM, compared to those with non-syndromic CFM, had a higher SDB score (0.27 ± 0.21 vs.0.21 ± 0.19, p = 0.003) and were more likely to have sleepiness (26% vs. 18%, p = 0.05).
  
  
  CONCLUSIONS
  Congenital craniofacial malformations in children are associated with high risk for SDB symptoms. Our findings should encourage a high index of suspicion for SDB in children with CFM, with a low threshold for further testing and close follow-up.
  
  
  BACKGROUND
  Moraleda-Cibrián M; Edwards SP; Kasten SJ; Berger M; Buchman SR; O'Brien LM. Symptoms of sleep disordered breathing in children with craniofacial malformations.
authors.affiliation: Sleep Disorders Center, Department of Neurology ; Department of Oral & Maxillofacial Surgery.
authors.forename: Marta
authors.initials: M
authors.lastname: Moraleda-Cibrián
authors.affiliation: Department of Oral & Maxillofacial Surgery.
authors.forename: Sean P
authors.initials: SP
authors.lastname: Edwards
authors.affiliation: Department of Surgery, Division of Plastic and Reconstructive Surgery, University of Michigan, Ann Arbor, MI.
authors.forename: Steven J
authors.initials: SJ
authors.lastname: Kasten
authors.affiliation: Department of Surgery, Division of Plastic and Reconstructive Surgery, University of Michigan, Ann Arbor, MI.
authors.forename: Mary
authors.initials: M
authors.lastname: Berger
authors.affiliation: Department of Surgery, Division of Plastic and Reconstructive Surgery, University of Michigan, Ann Arbor, MI.
authors.forename: Steven R
authors.initials: SR
authors.lastname: Buchman
authors.affiliation: Sleep Disorders Center, Department of Neurology ; Department of Oral & Maxillofacial Surgery.
authors.forename: Louise M
authors.initials: LM
authors.lastname: O'Brien
mesh_terms: D000293:Adolescent; D059327:Brachydactyly / Q000150:complications; D002648:Child; D002675:Child, Preschool; D019465:Craniofacial Abnormalities / Q000150:complications*; D003430:Cross-Sectional Studies; D004062:DiGeorge Syndrome / Q000150:complications; D006970:Disorders of Excessive Somnolence / Q000209:etiology; D019066:Facies; D005260:Female; D006801:Humans; D008607:Intellectual Disability / Q000150:complications; D008297:Male; D008342:Mandibulofacial Dysostosis / Q000150:complications; D010855:Pierre Robin Syndrome / Q000150:complications; D012891:Sleep Apnea Syndromes / Q000209:etiology*; D012913:Snoring / Q000209:etiology; D011795:Surveys and Questionnaires
keywords: Snoring; cleft; craniofacial anomalies; sleep-disordered breathing; sleepiness