Published online July 27, 2009
PEDIATRICS Vol. 124 No. 2 August 2009, pp. e313-e321 (doi:10.1542/peds.2008-3488)
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ARTICLE

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

Yona K. Cloonan, PhDa, Yemiserach Kifle, MDb,c, Scott Davis, PhDd,e, Matthew L. Speltz, PhDb,f, Martha M. Werler, ScDg and Jacqueline R. Starr, PhDb,c,d,e

a Department of Epidemiology, Michigan State University, East Lansing, Michigan
b Children's Craniofacial Center, Seattle Children's Hospital, Seattle, Washington; Departments of
c Pediatrics
d Epidemiology
f Psychiatry and Behavioral Sciences, University of Washington, Seattle, Washington
e Division of Public Health Sciences, Fred Hutchinson Cancer Research Center, Seattle, Washington
g Slone Epidemiology Center at Boston University, Boston, Massachusetts

OBJECTIVE: Children with craniofacial anomalies are at high risk for sleep-disordered breathing (SDB), yet its prevalence among children with craniofacial conditions is not known. Children with hemifacial microsomia (HFM) are likely particularly vulnerable to SDB as a result of 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 control subjects.

METHODS: We conducted a follow-up study among 124 case participants and 349 control subjects who previously participated in a study of HFM risk factors. 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 for control subjects (17%). Compared with control subjects, children with HFM more often had symptoms consistent with SDB. On average, case participants' parents reported 1.9 times as many symptoms on the PSQ breathing scale and 1.3 times more symptoms on the PSQ sleepiness scale than did control subjects' parents, with little difference on the PSQ behavior scale. Parents of children with HFM reported 1.4 times more night awakenings than did control subjects' parents.

CONCLUSIONS: Children with HFM experienced more snoring and other symptoms of SDB than did control subjects. Pediatricians should be aware of the increased vulnerability for SDB among children with mandibular or external ear underdevelopment or asymmetry and should refer to a sleep specialist as needed.

Key Words: hemifacial microsomia • oculoauriculovertebral syndrome • Goldenhar syndrome • craniofacial abnormalities • sleep • sleep-disordered breathing • snoring • child • epidemiology

Abbreviations: SDB—sleep-disordered breathing • HFM—hemifacial microsomia • PSQ—Pediatric Sleep Questionnaire • PSQ+—positive Pediatric Sleep Questionnaire screen for sleep-disordered breathing • AT—adenoidectomy or tonsillectomy • CI—confidence interval • aOR—adjusted odds ratio

Accepted Mar 30, 2009.


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