Sleep outcomes in children with single-suture craniosynostosis compared with unaffected controls

Public Health Significance\ud Craniosynostosis refers to premature closure of the cranial sutures during the perinatal period. Isolated single-suture craniosynostosis (SSC) is a non-syndromic form impacting only one suture. There is evidence that children with craniofacial anomalies have a higher risk for SDB as compared with unaffected children; however, it is not known whether this also applies to isolated SSC. Sleep-disordered breathing (SDB) occurs in 0.7% to 13.0% of children and is associated with behavioral, neurocognitive and cardiovascular consequences. We compared parent-reported SDB-related symptoms in children with and without isolated SSC, hypothesizing that those with SSC would have increased SDB-related symptoms as compared with controls. This preliminary study may have implications for clinical care by identifying a treatable condition (i.e. SDB) in an already vulnerable pediatric population.\ud Method\ud Among children with and without SSC, 6 SDB symptoms were assessed by parent report: restless sleep, arousal, snoring, snorting/gasping, apneas, and daytime sleepiness. Current sleep problems (yes/no) and ever sleep problems (yes/no) were identified based on the constellation of SDB-related outcomes. Snoring, the most common symptom of SDB, was categorized as never, 0-1 nights/week, and 2+ nights/week. Logistic and multinomial logistic regression models were applied to assess the association between sleep outcomes and SSC, adjusted for sex, age, race/ethnicity and family socioeconomic status (SES).\ud Results\ud Current sleep problems were reported slightly more often in children with SSC (19%) than in controls (14%; adjusted odds ratio (aOR)=1.6, 95% CI 0.9-2.8). There was no difference in report of having ever had sleep problems between cases and controls. In the multinomial regression model, the overall association of SSC and SDB was statistically significant (p=0.02). Snoring was more often reported as 2+ nights/week by parents of SSC cases (13%) than controls (4%) (versus never, aOR=3.4, 95% CI=1.4-7.9); while snoring 0-1 nights/week was reported similarly in both groups. \ud Conclusion\ud Children with SSC had significantly increased presence of snoring during sleep compared to controls. This suggests that children with isolated SSC may be at increased risk for SDB and further suggests that awareness and clinical evaluation of SDB are important for children with SSC. Further study using standardized assessments of SDB are needed.\u