TY - JOUR
T1 - Development and validation of a condition-specific quality of life instrument for adults with esophageal atresia
T2 - the SQEA questionnaire
AU - ten Kate, Chantal A.
AU - Teunissen, Nadine M.
AU - van Rosmalen, Joost
AU - Kamphuis, Lieke S.
AU - van Wijk, Michiel P.
AU - Joosten, Maja
AU - van Tuyll van Serooskerken, E. Sofie
AU - Wijnen, René
AU - IJsselstijn, Hanneke
AU - Rietman, André B.
AU - Spaander, Manon C. W.
N1 - Funding Information: We thank all participants for their time and effort filling out the questionnaires. We thank the Dutch patient association Vereniging voor Ouderen en Kinderen met een Slokdarmatresie (VOKS) for supporting this study and their input in the manuscript. We thank JoAnne Fruithof for her contribution in the expert team as a representative of the patient association, and Evelien Huizinga for her contribution in the expert team as an adult patient with esophageal atresia. We thank Michiel Luijten for his advice regarding item-response theory models. Publisher Copyright: © The Author(s) 2022. Published by Oxford University Press on behalf of International Society for Diseases of the Esophagus. All rights reserved.
PY - 2023/6/1
Y1 - 2023/6/1
N2 - The importance of multidisciplinary long-term follow-up for adults born with esophageal atresia (EA) is increasingly recognized. Hence, a valid, condition-specific instrument to measure health-related quality of life (HRQoL) becomes imperative. This study aimed to develop and validate such an instrument for adults with EA. The Specific Quality of life in Esophageal atresia Adults (SQEA) questionnaire was developed through focus group-based item generation, pilot testing, item reduction and a multicenter, nationwide field test to evaluate the feasibility, reliability (internal and retest) and validity (structural, construct, criterion and convergent), in compliance with the consensus-based standards for the selection of health measurement instruments guidelines. After pilot testing (n = 42), items were reduced from 144 to 36 questions. After field testing (n = 447), three items were discarded based on item-response theory results. The final SQEA questionnaire (33 items) forms a unidimensional scale generating an unweighted total score. Feasibility, internal reliability (Cronbach's alpha 0.94) and test-retest agreement (intra-class coefficient 0.92) were good. Construct validity was discriminative for esophageal replacement (P < 0.001), dysphagia (P < 0.001) and airway obstruction (P = 0.029). Criterion validity showed a good correlation with dysphagia (area under the receiver operating characteristic 0.736). SQEA scores correlated well with other validated disease-specific HRQoL scales such as the GIQLI and SGRQ, but poorly with the more generic RAND-36. Overall, this first condition-specific instrument for EA adults showed satisfactory feasibility, reliability and validity. Additionally, it shows discriminative ability to detect disease burden. Therefore, the SQEA questionnaire is both a valid instrument to assess the HRQoL in EA adults and an interesting signaling tool, enabling clinicians to recognize more severely affected patients.
AB - The importance of multidisciplinary long-term follow-up for adults born with esophageal atresia (EA) is increasingly recognized. Hence, a valid, condition-specific instrument to measure health-related quality of life (HRQoL) becomes imperative. This study aimed to develop and validate such an instrument for adults with EA. The Specific Quality of life in Esophageal atresia Adults (SQEA) questionnaire was developed through focus group-based item generation, pilot testing, item reduction and a multicenter, nationwide field test to evaluate the feasibility, reliability (internal and retest) and validity (structural, construct, criterion and convergent), in compliance with the consensus-based standards for the selection of health measurement instruments guidelines. After pilot testing (n = 42), items were reduced from 144 to 36 questions. After field testing (n = 447), three items were discarded based on item-response theory results. The final SQEA questionnaire (33 items) forms a unidimensional scale generating an unweighted total score. Feasibility, internal reliability (Cronbach's alpha 0.94) and test-retest agreement (intra-class coefficient 0.92) were good. Construct validity was discriminative for esophageal replacement (P < 0.001), dysphagia (P < 0.001) and airway obstruction (P = 0.029). Criterion validity showed a good correlation with dysphagia (area under the receiver operating characteristic 0.736). SQEA scores correlated well with other validated disease-specific HRQoL scales such as the GIQLI and SGRQ, but poorly with the more generic RAND-36. Overall, this first condition-specific instrument for EA adults showed satisfactory feasibility, reliability and validity. Additionally, it shows discriminative ability to detect disease burden. Therefore, the SQEA questionnaire is both a valid instrument to assess the HRQoL in EA adults and an interesting signaling tool, enabling clinicians to recognize more severely affected patients.
KW - esophageal atresia
KW - health-related quality of life
KW - quality of life
UR - http://www.scopus.com/inward/record.url?scp=85150787573&partnerID=8YFLogxK
U2 - https://doi.org/10.1093/dote/doac088
DO - https://doi.org/10.1093/dote/doac088
M3 - Article
C2 - 36562092
SN - 1120-8694
VL - 36
JO - Diseases of the esophagus : official journal of the International Society for Diseases of the Esophagus
JF - Diseases of the esophagus : official journal of the International Society for Diseases of the Esophagus
IS - 6
M1 - doac088
ER -