TY - JOUR
T1 - Establishment of a condition-specific quality-of-life questionnaire for children born with esophageal atresia aged 2–7 across 14 countries
AU - The International EA-QOL group
AU - Dellenmark-Blom, Michaela
AU - Witt, Stefanie
AU - Zendejas, Benjamin
AU - Sabolić, Ivana
AU - Porras-Hernandez, Juan Domingo
AU - Durkin, Natalie
AU - Eaton, Simon
AU - Birketvedt, Kjersti
AU - Sánchez Galán, Alba
AU - Eszter Müller, Katalin
AU - Rozensztrauch, Anna
AU - Soyer, Tutku
AU - Li, Siqi
AU - Fourtaka, Anastasia
AU - de Vos, Corne
AU - Slater, Graham
AU - Špoljarić, Ana
AU - Bennett, John
AU - Emblem, Ragnhild
AU - Andrásdi, Zita
AU - Smigiel, Robert
AU - Patkowski, Dariusz
AU - Durakbaşa, Çiğdem Ulukaya
AU - Stilinović, Marina
AU - Gottrand, Frederic
AU - Škrljak Šoša, Dora
AU - Luetić, Tomislav
AU - Gerus, Sylwester
AU - Yang, Shen
AU - Zhao, Yong
AU - Gu, Yichao
AU - Li, Shuangshuang
AU - Rodriguez-Alvirde, Diego
AU - Kadenczki, Orsolya
AU - Pasini, Miram
AU - Wallace, Vuokko
AU - Widenmann, Anke
AU - Milagres Sikwete, Feliciana
AU - Huang, Jinshi
AU - Martínez Martínez, Leopoldo
AU - Abrahamsson, Kate
AU - Izadi, Shawn
AU - Ure, Benno M
AU - Sidler, Daniel
AU - Quitmann, Julia H
AU - Dingemann, Jens
N1 - Publisher Copyright:
2023 The International EA-QOL Group.
PY - 2023/10/23
Y1 - 2023/10/23
N2 - Background: Esophageal atresia (EA) is a rare congenital anomaly characterized by a discontinuity of the esophagus. Following surgical repair, survival rates have improved dramatically the past decenniums and today exceed 90%, but the children commonly present with esophageal and respiratory morbidity. In 2018, a condition-specific quality-of-life questionnaire for children with esophageal atresia (EA) aged 2–7 in Sweden-Germany was finalized (The EA-QOL questionnaire). The study aim was to describe the evaluation of the new translations across 12 new countries in Europe, Asia, Africa, Central-and North America.Methods: Following forward-backward translation into the new languages, the 17-item EA-QOL questionnaire was tested in cognitive debriefing interviews with parents of children with EA aged 2–7. Parents rated if each item was easy to understand (clarity) and sensitive to answer (interference with personal integrity). They could skip responding to a non-applicable/problematic item and give open comments. Predefined psychometric criteria were used; item clarity ≥80%/item sensitive to answer ≤20%/item feasibility ≤5% missing item responses. The decision to modify the translation was based on native expert, patient stakeholder, and instrument developer review, and the need for harmonization between translations.Results: Similar to findings in the Swedish-German cognitive debriefing, the cross-cultural analysis of input from 116 parents from 12 new countries (4–14 parents, median 9 parents/country) showed that all items in the EA-QOL questionnaire fulfilled the criteria for item clarity ≥80% and sensitive to answer (ranging from 1%-4.5%), although results varied between countries. Four items had missing responses between 5.2% and 13.4%, three within the same domain and were in line with parents’ explanations. Poor translations and feasibility were improved.Conclusions: Based on parent input, the collaboration between native experts, patient stakeholders, and instrument developers, a linguistic version of the EA-QOL questionnaire for children aged 2–7 for use in and across 14 countries has been established. These efforts have set the conditions for a cross-cultural field test of the EA-QOL questionnaire and will open the doors for a new chapter in outcome research, registries, and clinical practice concerning children with EA. In the long-term, this will help increase knowledge of the disease's burden, promote patient-centeredness, exchange of information between nations, and strengthen evidence-based treatments for children born with EA.
AB - Background: Esophageal atresia (EA) is a rare congenital anomaly characterized by a discontinuity of the esophagus. Following surgical repair, survival rates have improved dramatically the past decenniums and today exceed 90%, but the children commonly present with esophageal and respiratory morbidity. In 2018, a condition-specific quality-of-life questionnaire for children with esophageal atresia (EA) aged 2–7 in Sweden-Germany was finalized (The EA-QOL questionnaire). The study aim was to describe the evaluation of the new translations across 12 new countries in Europe, Asia, Africa, Central-and North America.Methods: Following forward-backward translation into the new languages, the 17-item EA-QOL questionnaire was tested in cognitive debriefing interviews with parents of children with EA aged 2–7. Parents rated if each item was easy to understand (clarity) and sensitive to answer (interference with personal integrity). They could skip responding to a non-applicable/problematic item and give open comments. Predefined psychometric criteria were used; item clarity ≥80%/item sensitive to answer ≤20%/item feasibility ≤5% missing item responses. The decision to modify the translation was based on native expert, patient stakeholder, and instrument developer review, and the need for harmonization between translations.Results: Similar to findings in the Swedish-German cognitive debriefing, the cross-cultural analysis of input from 116 parents from 12 new countries (4–14 parents, median 9 parents/country) showed that all items in the EA-QOL questionnaire fulfilled the criteria for item clarity ≥80% and sensitive to answer (ranging from 1%-4.5%), although results varied between countries. Four items had missing responses between 5.2% and 13.4%, three within the same domain and were in line with parents’ explanations. Poor translations and feasibility were improved.Conclusions: Based on parent input, the collaboration between native experts, patient stakeholders, and instrument developers, a linguistic version of the EA-QOL questionnaire for children aged 2–7 for use in and across 14 countries has been established. These efforts have set the conditions for a cross-cultural field test of the EA-QOL questionnaire and will open the doors for a new chapter in outcome research, registries, and clinical practice concerning children with EA. In the long-term, this will help increase knowledge of the disease's burden, promote patient-centeredness, exchange of information between nations, and strengthen evidence-based treatments for children born with EA.
KW - children
KW - cognitive debriefing
KW - esophageal atresia
KW - quality of life
KW - rare disease
KW - translation
KW - validity
UR - http://www.scopus.com/inward/record.url?scp=85176149932&partnerID=8YFLogxK
U2 - 10.3389/fped.2023.1253892
DO - 10.3389/fped.2023.1253892
M3 - Article
SN - 2296-2360
VL - 11
JO - Frontiers in Pediatrics
JF - Frontiers in Pediatrics
M1 - 1253892
ER -