Obstructive sleep apnea in children with syndromic craniosynostosis: long-term respiratory outcome of midface advancement

Abstract Almost 50% of patients with Apert, Crouzon or Pfeiffer syndrome develop obstructive sleep apnea (OSA), mainly due to midface hypoplasia. Midface advancement is often the treatment of choice, but the few papers on long-term outcome report mixed results. This paper aimed to assess the long-te...

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Bibliographic Details
Published in:	International journal of oral and maxillofacial surgery 2010-02, Vol.39 (2), p.115-121
Main Authors:	Bannink, N, Nout, E, Wolvius, E.B, Hoeve, H.L.J, Joosten, K.F.M, Mathijssen, I.M.J
Format:	Article
Language:	eng
Subjects:	Acrocephalosyndactylia - complications Acrocephalosyndactylia - surgery Adolescent Airway Obstruction - etiology Biological and medical sciences Child Child, Preschool children Continuous Positive Airway Pressure Craniofacial Dysostosis - complications Craniofacial Dysostosis - surgery craniosynostosis Dentistry Diseases of the osteoarticular system Endoscopy Facial Bones - abnormalities Facial Bones - surgery Female Follow-Up Studies Humans Hypopharynx - physiopathology Longitudinal Studies Male Malformations and congenital and or hereditary diseases involving bones. Joint deformations Medical sciences midface advancement Nasopharyngeal Diseases - etiology obstructive sleep apnea Otorhinolaryngology. Stomatology Oxygen Inhalation Therapy Pharyngeal Diseases - etiology Pneumology Polysomnography Reconstructive Surgical Procedures - methods Respiration Respiratory Function Tests Respiratory system : syndromes and miscellaneous diseases Retrospective Studies Sleep Apnea, Obstructive - etiology Sleep Apnea, Obstructive - therapy Surgery Tomography, X-Ray Computed Tracheostomy Treatment Outcome Young Adult
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Summary:	Abstract Almost 50% of patients with Apert, Crouzon or Pfeiffer syndrome develop obstructive sleep apnea (OSA), mainly due to midface hypoplasia. Midface advancement is often the treatment of choice, but the few papers on long-term outcome report mixed results. This paper aimed to assess the long-term respiratory outcome of midface advancement in syndromic craniosynostosis with OSA and to determine factors contributing to its efficacy. A retrospective study was performed on 11 patients with moderate or severe OSA, requiring oxygen, continuous positive airway pressure (CPAP), or tracheostomy. Clinical symptoms, results of polysomnography, endoscopy and digital volume measurement of the upper airways on CT scan before and after midface advancement were reviewed. Midface advancement had a good respiratory outcome in the short term in 6 patients and was ineffective in 5. In all patients without respiratory effect or with relapse, endoscopy showed obstruction of the rhino- or hypopharynx. The volume measurements supported the clinical and endoscopic outcome. Despite midface advancement, long-term dependence on, or indication for, CPAP or tracheostomy was maintained in 5 of 11 patients. Pharyngeal collapse appeared to play a role in OSA. Endoscopy before midface advancement is recommended to identify airway obstruction that may interfere with respiratory improvement after midface advancement.
ISSN:	0901-5027 1399-0020