Introduction: Anatomic malformations associated with craniofacial abnormalities (CFAs) and skeletal dysplasias (SDs) contribute to airway obstruction during sleep in children, which may be treated surgically, with positive airway pressure (PAP) therapy, or with dental appliances. This study examines the management of OSA in children with CFAs and/or SDs, including the prevalence of residual OSA postsurgery and PAP therapy compliance.

Methods: This was a retrospective cohort study of children with CFAs and/or SD. Children were included if they underwent polysomnography (PSG) at the Queensland Children’s Hospital between December 2014 and April 2022.

Results: In this cohort of 150 children, 69% had a CFA, 25% had SD, and 5% had both a CFA and SD. The most common reason for referral to sleep clinic was snoring (61%). Half (n=75) received surgical intervention as first-line treatment (with adenotonsillectomy as the most common surgery), and of these children, 47 (77%) had no residual OSA, while 16 (23%) required further PAP therapy (as determined by PSG). PSG results were statistically similar between surgical and nonsurgical groups, but these data were skewed to more severe disease in the non-surgical group. PAP therapy adherence was 63% and was not age or sex dependent.

Conclusions: Surgical intervention was the most common first-line treatment offered to children with CFAs and/or SD. The rate of residual OSA following surgery was similar to previous reports, and CPAP was the most common form of PAP offered to children. Overall, PAP compliance in this cohort of children was also similar to previous reports.