Validation of a health-related quality of life instrument for primary ciliary dyskinesia (QOL-PCD)

Laura Behan, Margaret W. Leigh, Sharon D. Dell, Audrey Dunn Galvin, Alexandra Quittner, Jane S. Lucas

Research output: Contribution to journalArticle

14 Citations (Scopus)

Abstract

Background Quality of life (QOL)-primary ciliary dyskinesia (PCD) is the first disease-specific, health-related QOL instrument for PCD. Psychometric validation of QOL-PCD assesses the performance of this measure in adults, including its reliability, validity and responsiveness to change. Methods Seventy-Two adults (mean (range) age: 33â €..years (18-79â €..years); mean (range) FEV 1 % predicted: 68 (26-115)) with PCD completed the 49-item QOL-PCD and generic QOL measures: Short-Form 36 Health Survey, Sino-Nasal Outcome Test 20 (SNOT-20) and St George Respiratory Questionnaire (SGRQ)-C. Thirty-five participants repeated QOL-PCD 10-14â €..days later to measure stability or reproducibility of the measure. Results Multitrait analysis was used to evaluate how the items loaded on 10 hypothesised scales: physical, emotional, role and social functioning, treatment burden, vitality, health perceptions, upper respiratory symptoms, lower respiratory symptoms and ears and hearing symptoms. This analysis of item-To-Total correlations led to 9 items being dropped; the validated measure now comprises 40 items. Each scale had excellent internal consistency (Cronbach's α: 0.74 to 0.94). Two-week test-retest demonstrated stability for all scales (intraclass coefficients 0.73 to 0.96). Significant correlations were obtained between QOL-PCD scores and age and FEV 1. Strong relationships were also found between QOL-PCD scales and similar constructs on generic questionnaires, for example, lower respiratory symptoms and SGRQ-C (r=0.72, p<0.001), while weak correlations were found between measures of different constructs. Conclusions QOL-PCD has demonstrated good internal consistency, test-retest reliability, convergent and divergent validity. QOL-PCD offers a promising tool for evaluating new therapies and for measuring symptoms, functioning and QOL during routine care.

Original languageEnglish (US)
Pages (from-to)832-839
Number of pages8
JournalThorax
Volume72
Issue number9
DOIs
StatePublished - Sep 1 2017

Fingerprint

Kartagener Syndrome
Quality of Life
Reproducibility of Results
Health Surveys
Nose
Psychometrics
Hearing
Ear

Keywords

  • Psychology
  • Rare lung diseases
  • Respiratory Measurement

ASJC Scopus subject areas

  • Pulmonary and Respiratory Medicine

Cite this

Behan, L., Leigh, M. W., Dell, S. D., Dunn Galvin, A., Quittner, A., & Lucas, J. S. (2017). Validation of a health-related quality of life instrument for primary ciliary dyskinesia (QOL-PCD). Thorax, 72(9), 832-839. https://doi.org/10.1136/thoraxjnl-2016-209356

Validation of a health-related quality of life instrument for primary ciliary dyskinesia (QOL-PCD). / Behan, Laura; Leigh, Margaret W.; Dell, Sharon D.; Dunn Galvin, Audrey; Quittner, Alexandra; Lucas, Jane S.

In: Thorax, Vol. 72, No. 9, 01.09.2017, p. 832-839.

Research output: Contribution to journalArticle

Behan, L, Leigh, MW, Dell, SD, Dunn Galvin, A, Quittner, A & Lucas, JS 2017, 'Validation of a health-related quality of life instrument for primary ciliary dyskinesia (QOL-PCD)', Thorax, vol. 72, no. 9, pp. 832-839. https://doi.org/10.1136/thoraxjnl-2016-209356
Behan, Laura ; Leigh, Margaret W. ; Dell, Sharon D. ; Dunn Galvin, Audrey ; Quittner, Alexandra ; Lucas, Jane S. / Validation of a health-related quality of life instrument for primary ciliary dyskinesia (QOL-PCD). In: Thorax. 2017 ; Vol. 72, No. 9. pp. 832-839.
@article{565dd2ea76a640b28ef5ef8286dbd9d8,
title = "Validation of a health-related quality of life instrument for primary ciliary dyskinesia (QOL-PCD)",
abstract = "Background Quality of life (QOL)-primary ciliary dyskinesia (PCD) is the first disease-specific, health-related QOL instrument for PCD. Psychometric validation of QOL-PCD assesses the performance of this measure in adults, including its reliability, validity and responsiveness to change. Methods Seventy-Two adults (mean (range) age: 33{\^a} €..years (18-79{\^a} €..years); mean (range) FEV 1 {\%} predicted: 68 (26-115)) with PCD completed the 49-item QOL-PCD and generic QOL measures: Short-Form 36 Health Survey, Sino-Nasal Outcome Test 20 (SNOT-20) and St George Respiratory Questionnaire (SGRQ)-C. Thirty-five participants repeated QOL-PCD 10-14{\^a} €..days later to measure stability or reproducibility of the measure. Results Multitrait analysis was used to evaluate how the items loaded on 10 hypothesised scales: physical, emotional, role and social functioning, treatment burden, vitality, health perceptions, upper respiratory symptoms, lower respiratory symptoms and ears and hearing symptoms. This analysis of item-To-Total correlations led to 9 items being dropped; the validated measure now comprises 40 items. Each scale had excellent internal consistency (Cronbach's α: 0.74 to 0.94). Two-week test-retest demonstrated stability for all scales (intraclass coefficients 0.73 to 0.96). Significant correlations were obtained between QOL-PCD scores and age and FEV 1. Strong relationships were also found between QOL-PCD scales and similar constructs on generic questionnaires, for example, lower respiratory symptoms and SGRQ-C (r=0.72, p<0.001), while weak correlations were found between measures of different constructs. Conclusions QOL-PCD has demonstrated good internal consistency, test-retest reliability, convergent and divergent validity. QOL-PCD offers a promising tool for evaluating new therapies and for measuring symptoms, functioning and QOL during routine care.",
keywords = "Psychology, Rare lung diseases, Respiratory Measurement",
author = "Laura Behan and Leigh, {Margaret W.} and Dell, {Sharon D.} and {Dunn Galvin}, Audrey and Alexandra Quittner and Lucas, {Jane S.}",
year = "2017",
month = "9",
day = "1",
doi = "10.1136/thoraxjnl-2016-209356",
language = "English (US)",
volume = "72",
pages = "832--839",
journal = "Thorax",
issn = "0040-6376",
publisher = "BMJ Publishing Group",
number = "9",

}

TY - JOUR

T1 - Validation of a health-related quality of life instrument for primary ciliary dyskinesia (QOL-PCD)

AU - Behan, Laura

AU - Leigh, Margaret W.

AU - Dell, Sharon D.

AU - Dunn Galvin, Audrey

AU - Quittner, Alexandra

AU - Lucas, Jane S.

PY - 2017/9/1

Y1 - 2017/9/1

N2 - Background Quality of life (QOL)-primary ciliary dyskinesia (PCD) is the first disease-specific, health-related QOL instrument for PCD. Psychometric validation of QOL-PCD assesses the performance of this measure in adults, including its reliability, validity and responsiveness to change. Methods Seventy-Two adults (mean (range) age: 33â €..years (18-79â €..years); mean (range) FEV 1 % predicted: 68 (26-115)) with PCD completed the 49-item QOL-PCD and generic QOL measures: Short-Form 36 Health Survey, Sino-Nasal Outcome Test 20 (SNOT-20) and St George Respiratory Questionnaire (SGRQ)-C. Thirty-five participants repeated QOL-PCD 10-14â €..days later to measure stability or reproducibility of the measure. Results Multitrait analysis was used to evaluate how the items loaded on 10 hypothesised scales: physical, emotional, role and social functioning, treatment burden, vitality, health perceptions, upper respiratory symptoms, lower respiratory symptoms and ears and hearing symptoms. This analysis of item-To-Total correlations led to 9 items being dropped; the validated measure now comprises 40 items. Each scale had excellent internal consistency (Cronbach's α: 0.74 to 0.94). Two-week test-retest demonstrated stability for all scales (intraclass coefficients 0.73 to 0.96). Significant correlations were obtained between QOL-PCD scores and age and FEV 1. Strong relationships were also found between QOL-PCD scales and similar constructs on generic questionnaires, for example, lower respiratory symptoms and SGRQ-C (r=0.72, p<0.001), while weak correlations were found between measures of different constructs. Conclusions QOL-PCD has demonstrated good internal consistency, test-retest reliability, convergent and divergent validity. QOL-PCD offers a promising tool for evaluating new therapies and for measuring symptoms, functioning and QOL during routine care.

AB - Background Quality of life (QOL)-primary ciliary dyskinesia (PCD) is the first disease-specific, health-related QOL instrument for PCD. Psychometric validation of QOL-PCD assesses the performance of this measure in adults, including its reliability, validity and responsiveness to change. Methods Seventy-Two adults (mean (range) age: 33â €..years (18-79â €..years); mean (range) FEV 1 % predicted: 68 (26-115)) with PCD completed the 49-item QOL-PCD and generic QOL measures: Short-Form 36 Health Survey, Sino-Nasal Outcome Test 20 (SNOT-20) and St George Respiratory Questionnaire (SGRQ)-C. Thirty-five participants repeated QOL-PCD 10-14â €..days later to measure stability or reproducibility of the measure. Results Multitrait analysis was used to evaluate how the items loaded on 10 hypothesised scales: physical, emotional, role and social functioning, treatment burden, vitality, health perceptions, upper respiratory symptoms, lower respiratory symptoms and ears and hearing symptoms. This analysis of item-To-Total correlations led to 9 items being dropped; the validated measure now comprises 40 items. Each scale had excellent internal consistency (Cronbach's α: 0.74 to 0.94). Two-week test-retest demonstrated stability for all scales (intraclass coefficients 0.73 to 0.96). Significant correlations were obtained between QOL-PCD scores and age and FEV 1. Strong relationships were also found between QOL-PCD scales and similar constructs on generic questionnaires, for example, lower respiratory symptoms and SGRQ-C (r=0.72, p<0.001), while weak correlations were found between measures of different constructs. Conclusions QOL-PCD has demonstrated good internal consistency, test-retest reliability, convergent and divergent validity. QOL-PCD offers a promising tool for evaluating new therapies and for measuring symptoms, functioning and QOL during routine care.

KW - Psychology

KW - Rare lung diseases

KW - Respiratory Measurement

UR - http://www.scopus.com/inward/record.url?scp=85020514939&partnerID=8YFLogxK

UR - http://www.scopus.com/inward/citedby.url?scp=85020514939&partnerID=8YFLogxK

U2 - 10.1136/thoraxjnl-2016-209356

DO - 10.1136/thoraxjnl-2016-209356

M3 - Article

C2 - 28246220

AN - SCOPUS:85020514939

VL - 72

SP - 832

EP - 839

JO - Thorax

JF - Thorax

SN - 0040-6376

IS - 9

ER -