Reliability and Validity of the Obstructive Sleep Apnea-18 Survey in Healthy Children and Children With Syndromic Craniosynostosis

Abstract

Obstructive sleep apnea (OSA) affects a person's quality of life. A questionnaire, the OSA-18, is available to measure quality of life in children with OSA not caused by specific craniofacial syndromes. We assessed the internal consistency, test-retest reliability, and discriminative validity of the OSA-18 in children with syndromic and complex craniosynostosis; we also applied the OSA-18 in healthy children to obtain reference values. The OSA-18 was translated in the Dutch language using the procedure of multiple forward and backward translations. Test-retest reliability and internal consistency were examined. In a prospective study, the craniosynostosis patients underwent an ambulatory polysomnography to diagnose OSA. The ability of the OSA-18 to discriminate between subgroups of patients with or without OSA was evaluated. We compared OSA-18 scores of children with syndromic or complex craniosynostosis with scores in healthy children. The Cronbach's alpha was ≥ 0.70 for the total OSA-18 score and for most of the domains in both the craniosynostosis and general population. In the craniosynostosis group, the test-retest intraclass correlation coefficients were ≥ 0.70, except for the domain physical suffering at 0.69. The discriminative validity of the domains sleep disturbance, physical suffering, caregiver concerns, and total OSA-18 score was significant between the general and craniosynostosis population. This study supports the reliability and validity of the OSA-18 in children with syndromic or complex craniosynostosis.