Psychometric Properties and Predictive Value of a Screening Questionnaire for Obstructive Sleep Apnea in Young Children With Down Syndrome.

Sarah Grantham-Hill,Catherine Tuffrey,Ruth N Kingshott,Anna Joyce,Hazel J Evans,Paul Gringras,Jane Martin,Catherine M Hill,Emma Sanders,Janine Reynolds,Heather E Elphick,Karen Spruyt

doi:10.3389/fpsyt.2020.00285

Abstract

Study ObjectivesObstructive sleep apnea (OSA) is common in children with Down syndrome (DS) and is associated with adverse health and cognitive outcomes. Daytime clinical assessment is poorly predictive of OSA, so regular screening with sleep studies is recommended. However, sleep studies are costly and not available to all children worldwide. We aimed to evaluate the psychometric properties and predictive value of a newly developed screening questionnaire for OSA in this population.Methods202 children aged 6 months to 6th birthday with DS were recruited, of whom 188 completed cardio-respiratory sleep studies to generate an obstructive apnea hypopnea index (OAHI). Parents completed the 14-item Down syndrome OSA screening questionnaire. Responses were screened, a factor analysis undertaken, internal consistency calculated and receiver operator characteristic (ROC) curves drawn to generate an area under the curve (AUC) to assess criterion related validity.ResultsOf 188 children who completed cardiorespiratory sleep studies; parents completed the screening questionnaire for 186. Of this study population 15.4% had moderate to severe OSA defined by an OAHI of ≥5/h. Sixty-three (33.9%) participants were excluded due to “unsure” responses or where questions were not answered. Using the remaining 123 questionnaires a four-factor solution was found, with the 1st factor representing breathing related symptoms, explaining a high proportion of the variance. Internal consistency was acceptable with a Cronbach alpha of 0.87. ROC curves for the total score generated an AUC statistic of 0.497 and for the breathing subscale an AUC of 0.603 for moderate to severe OSA.ConclusionA well designed questionnaire with good psychometric properties had limited predictive value to screen for moderate to severe OSA in young children with DS. The use of a screening questionnaire is not recommended. Screening for OSA in this population requires objective sleep study measures.

Highlights

Down syndrome (DS) is the commonest chromosomal abnormality affecting approximately 1:1,200 live births worldwide [1]
The present study aimed to evaluate the psychometric properties and predictive value of this questionnaire when tested in a population of young children with DS
Two hundred two participants enrolled in the study, of whom 186 had both a completed DS obstructive sleep apnea (OSA) questionnaire and a calculated obstructive apnea/hypopnea index (OAHI)

Summary

Introduction

Down syndrome (DS) is the commonest chromosomal abnormality affecting approximately 1:1,200 live births worldwide [1]. OSA is estimated to affect 75%, of this population compared to 1.2% of typically developing (TD) children [2, 3]. OSA causes nocturnal hypoxia and fragmented sleep with adverse health consequence that have been extensively studied in TD children including: hypertension (systemic and pulmonary) [5], cognitive deficits (impaired attention and executive function) leading to impaired learning and school performance [6], as well as reduced quality of life [7], and increased health care utilization [8]. We have recently reported that OSA predicts deficits in parent-reported executive function behaviors in very young children with Down syndrome [11]. It has further been hypothesized that OSA in DS may be a risk factor for the development of Alzheimer’s disease [12]. Prompt identification and treatment of OSA in DS is an important goal

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