Sleep Outcomes in Children With Hemifacial Microsomia and Controls: A Follow-up Study

Abstract

Objective-Children with craniofacial anomalies are at high risk of sleep-disordered breathing (SDB), yet its prevalence among children with craniofacial conditions is not known. Children with a relatively common congenital craniofacial condition, hemifacial microsomia (HFM), are likely particularly vulnerable to SDB due to underdevelopment of the mandible and oropharynx. Nevertheless, most children with HFM are not referred for sleep studies. We hypothesized that sleep outcomes would be worse in children with HFM versus controls.Methods-We conducted a follow-up study among 124 cases and 349 controls who previously participated in a study of HFM risk factors. Cases were eligible if diagnosed by a craniofacial specialist at one of 25 participating centers. Controls were matched to cases by pediatric practice and age at interview. Parents completed the Pediatric Sleep Questionnaire (PSQ) regarding symptoms of SDB and sleep habits. Regression models were adjusted for region, age, sex, race/ethnicity and maternal education. Results-Snoring was more commonly reported for children with HFM (29%) than controls (17%)[adjusted odds ratio (adjOR)=1.9, 95% confidence interval (CI) 1.1-3.2]. Compared with controls, children with HFM more often had symptoms consistent with SDB (adjOR=2.8, CI 1.5-5.1). On average, cases' parents reported 1.9 times as many symptoms on the PSQ breathing scale (CI 1.4-2.7), and 1.3 times more symptoms on the PSQ sleepiness scale (CI 0.9-1.8) than did controls' parents, with little difference on the PSQ behavior scale. Parents of children with HFM reported 1.4 times more night awakenings than did controls' parents (CI 0.9-2.2). There was no association for sleep-onset latency or time in bed.Conclusions-Children with HFM experienced more snoring and other symptoms of SDB than controls. Pediatricians should be aware of the increased vulnerability for SDB among children with

Obstructive sleep apnoea in children with craniofacial syndromes

“…One study found this population to have increased symptoms of breathing difficulties during sleep, sleepiness, and night awakenings compared to controls 28 . Another study found that patients with hemifacial microsomia who have more severe mandibular deformities were at higher risk for OSAS 29 .…”

Health-Related Quality of Life in Children with Hemifacial Microsomia

“…14,17,18 We have shown that HFM-affected children are 2 to 3 times as likely to score in the at-risk range for neurocognitive and psychosocial problems with receptive language and visual-motor impairments 18 and have less social competence and peer acceptance per teacher report. 14 In addition to assessments of the child’s neuropsychological performance, social competencies and behavior, and sleep patterns, we also gathered information from children and their parents about the child’s HRQOL, that is, the primary focus of this study.…”

The particular case of sleep-disordered breathing in syndromic patients

“…2). A casecontrol study in children over 3 years of age with hemifacial microsomia treated in 26 American and Canadian craniofacial centers between 1993 and 2002 found significantly higher rates of snoring and sleep-disordered breathing in children with hemifacial microsomia than in controls 4 . OSAS is also an element in the clinical presentation associated with Prader-Willior Rohhadnet syndrome, for which polysomnography should be systematic 11 .…”