Agreement between mothers’, fathers’, and children’s’ ratings on health-related quality of life in children born with esophageal atresia – a German cross-sectional study

Abstract

Background Esophageal atresia (EA) is a rare congenital malformation, which is characterized by the discontinuity of the esophagus. We investigated the agreement between mothers’, fathers’, and children’s’ ratings on health-related quality of life (HRQOL) in children born with EA. We aimed to broaden the understanding of subjective experiences of HRQOL from different perspectives. We hypothesized that the agreement between mother and father ratings would be high, whereas the agreement between child and mother ratings as well as child and father ratings would show more substantial differences. Methods We obtained data from 40 families (23 mother-father dyads of children aged 2–7 years and 17 mother-father-child triads of children and adolescents aged 8–18 years) with children born with EA, who were treated in two German hospitals. HRQOL was measured using the generic PedsQL™ questionnaires and the condition-specific EA-QOL© questionnaires. We calculated intraclass coefficients and performed one-way repeated measures ANOVAs to analyze differences for each domain as well as for the total scores. Results Intraclass correlation coefficients (ICCs) indicated a strong agreement (≥.80) between mother and father reports of children’s HRQOL for both generic and condition-specific measurements. The ICCs for the generic HRQOL for mother/father-child-dyads revealed only fair to good agreement, whereas ICCs for condition-specific HRQOL showed high agreement for mother-child and father-child-agreement. Analyses of Covariance revealed differences in mother/father-child agreement in the generic domain School, both parents reporting lower HRQOL scores than the children themselves. Fathers reported significantly higher scores in the condition-specific domain Social than their children. Conclusions Results showed that mothers’ and fathers’ reports corresponded to each other. Nonetheless, these reports might not be interchangeably used because mother-child and father-child agreement showed differences. Children might know the best on how they feel, and parent proxy-report is recommended when reasons such as young age, illness, or cognitive impairments do not allow to ask the child. But parent-report – no matter if reported by mother or father – should only be an additional source to broaden the view on the child’s health status and well-being. The current study contributes to a better understanding of the complex family relationships involved when parenting a child born with EA.

Health-Related Quality of Life in Patients after Repair of Esophageal Atresia: A Review of Current Literature

“…described HRQoL among children and adolescents born with EA, [24][25][26][27][28][29][30][31][32][33][34][35][36][37][38] three of which 32,34,38 also reported on HRQoL among adults (patients >18 years old). The remaining two articles described HRQoL among adults with EA only.…”

“…Small sample sizes motivate investigators to mix children's and parents' HRQoL scores/ratings, 25 which is not appropriate from the methodological perspective, as the views of EA children should be regarded as a primary source of their HRQoL. 37 Several studies are single-center studies, lacking matched controls, and raw data from general samples, which impede the statistical investigations of HRQoL in patients with EA. Some studies also suffer from a large proportion of patients who were lost to follow-up or have low response rates to the HRQoL questionnaires (►Table 1).…”

“…Eight studies of pediatric patients recruited families from the same Swedish and/or German center cohorts. 24,26,[29][30][31][35][36][37] Factors that are investigated as having an influence on HRQoL in patients with EA vary by definition, and there is an emphasis on clinical variables. As stated by Tan Tanny et al, 53 specific HRQoL assessments in patients with LGEA are needed.…”

Care Recommendations for the Respiratory Complications of Esophageal Atresia-Tracheoesophageal Fistula: The International Network of Esophageal Atresia, Respiratory Complications Working Group

“…Some centres have also included diagnostic and interventional radiologists and an intensivist 156 . Special emphasis is given to have regular evaluation and follow-up by psychologists because of growing concern about the long-term effect of EA-TEF on the quality of life of the patients and their families [157][158][159][160] .…”

Health-Related Quality of Life Assessment in Children and their Families: Aspects of Importance to the Pediatric Surgeon

“…The authors further concluded that congruence in HRQOL assessment of children with rare chronic diseases is important for the long-term well-being of families since discrepancies might have clinical implications regarding the focus of the treatment. 45 Furthermore, since often only one parent is present in pediatric settings, knowing about possible discrepancies in HRQOL ratings between mothers and fathers is important to interpret HRQOL rankings correctly.…”