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

Abstract

Study Objectives: Obstructive 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.Methods: 202 children aged 6 months to 6 th 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.Results: Of 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 1 st 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

Detecting sleep apnea in adults with Down syndrome using WatchPAT: A feasibility study

“…Our study revealed a gap between subjective STOP-Bang scores and objective WatchPAT measurements. We showed that informant-based questionnaires (i.e., STOP-Bang) did not reflect sleep disturbances in this population, resembling previous findings (Giménez et al, 2018;Grantham-Hill et al, 2020;Tadokoro et al, 2020). This is potentially related to the fact that the questionnaire reflects the caregivers' perceptions, but caregivers are generally not present at night in the bedroom (which is different from a partner), making it difficult for them to answer questions regarding, for example, snoring.…”

Prediction of obstructive sleep apnoea in children and adolescents with Down syndrome

“…However, we found that many questionnaire outcomes including OSA-related QOL, behaviour, daytime functioning and problems related to sleep including sleep routine, bedtime anxiety, morning tiredness, night arousal and restless sleep were not related to the OAHI. Many studies have identified the poor predictive qualities of questionnaires for the presence of OSA in TD children and adolescents as well as those with other conditions (Borgstrom et al 2013;Pabary et al 2016;Walter et al 2016;Grantham-Hill et al 2020), and in children and adolescents with DS there is the additional issue that OSA symptoms may be attributed to the neurocognitive and behavioural impacts of DS itself. In recognition of this, one study published after the commencement of our study designed the DS-OSA questionnaire to better capture OSA-specific symptomology in children with DS (Grantham-Hill et al 2020), but found poor predictive qualities for this tool.…”

“…Considering the challenges of accessing PSG services worldwide, some studies have focused on using screening questionnaires to identify significant OSA in children and adolescents with DS. However, the utility of the tools as clinical indicators for moderate-severe OSA in this population has not been supported particularly due to poor positive predictive values (PPVs) (Pabary et al 2016;Grantham-Hill et al 2020).…”

Behavioural and emotional disturbances associated with sleep‐disordered breathing symptomatology in children with Down's syndrome

“…Both the PaedESS and ESS scores were significantly positively correlated with the TBPS in the univariate regression analysis, but not in the multivariate model, and were higher in children with symptoms suggestive of SDB. With respect to limitations, we used surrogate markers of SDB along with assessing subjective sleepiness – both insufficiently sensitive for SDB in children with DS, as shown in subsequent studies (Maris, Verhulst, Wojciechowski, Van de Heyning, & Boudewyns, 2016; Lee et al, 2018; Grantham‐Hill et al, 2020). Other factors, such as settling problems, were not accounted for (Carter, McCaughey, Annaz, & Hill, 2009).…”