The Esophageal-Atresia-Quality-of-life Questionnaires: Feasibility, Validity and Reliability in Sweden and Germany

• Published in: Journal of pediatric gastroenterology and nutrition Vol. 67; no. 4; pp. 469 - 477
• Main Authors: Dellenmark-Blom, Michaela, Dingemann, Jens, Witt, Stefanie, Quitmann, Julia H, Jönsson, Linus, Gatzinsky, Vladimir, Chaplin, John E, Bullinger, Monika, Flieder, Sofie, Ure, Benno M, Dingemann, Carmen, Abrahamsson, Kate
• Format: Journal Article
• Language: English
• Published: United States by European Society for Pediatric Gastroenterology, Hepatology, and Nutrition and North American Society for Pediatric Gastroenterology 01.10.2018
• Subjects: Kirurgi; Surgery
• ISSN: 0277-2116; 1536-4801; 1536-4801
• DOI: 10.1097/MPG.0000000000002019

OBJECTIVES:Esophageal atresia (EA) is a rare malformation characterized of discontinuity of the esophagus, concurrent with or without a tracheoesophageal fistula (TEF). We report the feasibility validity and reliability of a condition-specific quality-of-life (QOL) tool for EA/TEF children, the age-adapted EA-QOL-questionnaires, when used in Sweden and Germany. METHODS:A total of 124 families of children with EA/TEF participated in the study; 53 parents completed the EA-QOL-questionnaire for children aged 2 to 7 years; 62 children/71 parents the EA-QOL-questionnaire for children 8 to 17 years. Feasibility was determined from the percentage of missing item responses. Based on clinical data and previously validated generic QOL-instruments (PedsQL 4.0, DISABKIDS-12), the final EA-QOL scores were evaluated against hypotheses of validity (known-groups/concurrent/convergent) and reliability (internal consistency/retest reliability of scores for 3 weeks). Significant level was P < 0.05. RESULTS:In the questionnaire for EA/TEF children aged 2 to 7 years, 16/18 items were completed with missing values <6% (range 0%–7.5%), and in the questionnaire for 8 to 17-year-olds, 24/24 child-reported items (range 0%–4.8%) and 21/24 parent-reported items (range 0%–7.0%). In both age-specific EA-QOL-questionnaires, desirable standards for known-groups and concurrent validity were fulfilled; digestive symptoms and feeding difficulties negatively impacted EA-QOL-Total-scores (P < 0.001), and as hypothesized, in 2 to 7-year-olds, respiratory symptoms decreased EA-QOL-Total-scores (P = 0.002). Correlations between the EA-QOL and generic QOL questionnaires supported convergent validity. Internal consistency reliability was satisfactory. The level of agreements of EA-QOL-scores between the field- and retest study were good to excellent. CONCLUSIONS:The overall psychometric performance of the EA-QOL-questionnaires for EA/TEF children is satisfactory and can enhance outcome evaluations in future research and clinical practice.