Factors of family impact in a Swedish–German cohort of children born with esophageal atresia

Abstract

Background After repair of esophageal atresia (EA), childhood survivors commonly present with digestive and respiratory morbidity, and around 55% have associated anomalies. Although it is known that these problems can reduce health-related quality of life in children with EA, less is understood about the impact on the family. We aimed to identify factors related to family impact in children with EA. Methods One parent each of a child with EA (2–18 years) in 180 families from Sweden and Germany answered the PedsQL™ Family Impact Module as the dependent variable. The independent variables were the child’s parent-reported health-related quality of life as measured by PedsQL™ 4.0, current symptoms, school situation, and parent/family characteristics together with child clinical data from the medical records. Results Stepwise multivariable regression analysis showed a multifactorial model of the total family impact scores (R2 = 0.60), with independent factors being the child’s overall generic health-related quality of life, school-absence ≥ 1/month, severe tracheomalacia, a family receiving carer’s allowance, and a parent with no university/college education, p < 0.05. Logistic regression analysis showed that an increased number of symptoms in the child the preceding 4 weeks lowered the family impact scores; however, the child’s feeding (R2 = 0.35) and digestive symptoms (R2 = 0.25) explained more in the variation of scores than the child’s respiratory symptoms (R2 = 0.09), p < 0.0001. Conclusions Family functioning may be a contributing factor to the maintenance of child health. The study findings suggest multifactorial explanations to family impact in children with EA, which are essential when optimizing the support to these families in clinical and psychosocial practice. Future research should explore experiences of family impact from all family members’ perspectives and multicenter studies are warranted to understand better the effectiveness of psychosocial-educational interventions to families of children with EA.

A Narrative Review of Patient-Reported Outcome Measures and Their Application in Recent Pediatric Surgical Research: Advancing Knowledge and Offering New Perspectives to the Field

“…Out of 345 articles identified by the database searches, 49 articles met the inclusion criteria. 28 29 30 31 32 33 34 35 36 37 38 39 40 41 42 43 44 45 46 47 48 49 50 51 52 53 54 55 56 57 58 59 60 61 62 63 64 65 66 67 68 69 70 71 72 73 74 75 76 Most concerned esophageal atresia ( n = 17) and Hirschsprung's disease ( n = 14). None of the articles focused on duodenal atresia or omphalocele.…”

Experiences of schooling in children with long-gap esophageal atresia in Sweden

“…Parents' open answers were categorized according to their content; type of school support and reason for school absence and then analyzed using descriptive statistics. School absence ≥ 1 times/month the past year was regarded as high frequency [19,34]. For continuous variables, median and range were calculated and for categorical variables, frequencies and percentages.…”

“…Respiratory morbidity is very common at young age [25,39] and in this study school absence also correlated to young child age, suggesting an interrelationship. Knowledge of school absence in children with LGEA is important to recognize, as it has been described by parents of children with EA to impair social integration with peers and school achievement [19] and it is an independent factor that negatively in uences family functioning [34].…”

“…However, given the small sample size despite our nationwide recruitment of children with LGEA, investigations into narrower age subgroups would not be statistically feasible. Moreover, there is no standardized de nition of problematic school absence, and we de ned a cutoff for high frequency of school absence which had been previously used [19,34]. Although we complied with terminology utilized by the Swedish National Agency for Education[28], the school questions, apart from those in the PedsQL 4.0, were not part of a valid scoring instrument and we collected data from parents.…”

Psychometric properties of the European Portuguese version of the Pediatric Quality of Life Inventory™ family impact module

“…This scale has been used internationally to assess health‐related quality of life in families of children with a diversity of chronic diseases, namely epilepsy (Rozensztrauch & Kołtuniuk, 2022), esophageal atresia (Dellenmark‐Blom et al, 2022), nephrotic syndrome (Mishra et al, 2015), drepanocytosis (Panepinto et al, 2009), and Rett syndrome (Rozensztrauch et al, 2021). Its psychometric properties have been positively evaluated in all these studies, as well in another undertaken with families of healthy children, ensuring its reliability and validity (Medrano et al, 2013).…”