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Psychometric properties and predictive value of a screening questionnaire for obstructive sleep apnea in young children with Down syndrome

Psychometric properties and predictive value of a screening questionnaire for obstructive sleep apnea in young children with Down syndrome
Psychometric properties and predictive value of a screening questionnaire for obstructive sleep apnea in young children with Down syndrome

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 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. 

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 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. 

Conclusion: a 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.

Down syndrome, obstructive sleep apnea/apnea, psychometric properties, screening, trisomy 21
1664-0640
Grantham-Hill, Sarah
e0193e37-4905-4e83-b4fa-c505f8d28fdc
Evans, Hazel J.
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Tuffrey, Catherine
04791b74-8462-41bd-9207-c0eb64dd56ff
Sanders, Emma
0765a840-7392-4f28-938b-217ef3f1653b
Elphick, Heather E.
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Gringras, Paul
fdb21ba5-b96f-453d-849c-6a6827ddf6cd
Kingshott, Ruth N.
d93170f2-b232-4296-b5af-cd2856f1f64a
Martin, Jane
2111f95f-14ac-4ca6-a70a-42fdda5aede8
Reynolds, Janine
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Joyce, Anna
b0a0c3c1-0792-4d2a-a50b-251cae879884
Hill, Catherine M.
172dc851-574e-4815-9c5e-4de4dae9fc35
Spruyt, Karen
bdc725a4-4c8e-40d9-b7ed-f9477888abf1
Grantham-Hill, Sarah
e0193e37-4905-4e83-b4fa-c505f8d28fdc
Evans, Hazel J.
11506bb3-ce57-41fa-9966-0b22131a1a35
Tuffrey, Catherine
04791b74-8462-41bd-9207-c0eb64dd56ff
Sanders, Emma
0765a840-7392-4f28-938b-217ef3f1653b
Elphick, Heather E.
35d656dc-594d-497c-ac14-aa62565e4cce
Gringras, Paul
fdb21ba5-b96f-453d-849c-6a6827ddf6cd
Kingshott, Ruth N.
d93170f2-b232-4296-b5af-cd2856f1f64a
Martin, Jane
2111f95f-14ac-4ca6-a70a-42fdda5aede8
Reynolds, Janine
1ee71ed2-0a70-4b58-b043-77853eccb769
Joyce, Anna
b0a0c3c1-0792-4d2a-a50b-251cae879884
Hill, Catherine M.
172dc851-574e-4815-9c5e-4de4dae9fc35
Spruyt, Karen
bdc725a4-4c8e-40d9-b7ed-f9477888abf1

Grantham-Hill, Sarah, Evans, Hazel J., Tuffrey, Catherine, Sanders, Emma, Elphick, Heather E., Gringras, Paul, Kingshott, Ruth N., Martin, Jane, Reynolds, Janine, Joyce, Anna, Hill, Catherine M. and Spruyt, Karen (2020) Psychometric properties and predictive value of a screening questionnaire for obstructive sleep apnea in young children with Down syndrome. Frontiers in Psychiatry, 11, [285]. (doi:10.3389/fpsyt.2020.00285).

Record type: Article

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 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. 

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 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. 

Conclusion: a 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.

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Accepted/In Press date: 24 March 2020
e-pub ahead of print date: 28 April 2020
Published date: 28 April 2020
Additional Information: Funding Information: this study was supported by Action Medical Research and the Garfield Weston Foundation [grant reference 2040]. We would like to thank the UK Down Syndrome Medical Interest group as well as the Down Syndrome Association for their help with recruiting children to the study. Most importantly we thank the children and families for their enthusiasm to take part. The authors would also like to acknowledge the Southampton NIHR Wellcome Trust Clinical Research Facility for their support of this work.
Keywords: Down syndrome, obstructive sleep apnea/apnea, psychometric properties, screening, trisomy 21

Identifiers

Local EPrints ID: 439196
URI: http://eprints.soton.ac.uk/id/eprint/439196
ISSN: 1664-0640
PURE UUID: 8efd5a66-a125-4e89-969a-f678e5dadf24
ORCID for Hazel J. Evans: ORCID iD orcid.org/0000-0001-9366-556X

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Date deposited: 06 Apr 2020 16:36
Last modified: 18 Mar 2024 03:46

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Contributors

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

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