Parent–child-agreement on health-related quality of life and its determinants in patients born with Esophageal Atresia: a Swedish–German cross-sectional study

Orphanet Journal of Rare Diseases

Stefanie Witt¹  Julia Hannah Quitmann¹  Susanne Kuckuck¹  Monika Bullinger¹  Benno Ure²  Carmen Dingemann²  Jens Dingemann²  Vladimir Gatzinsky³  Linus Jönsson³  Michaela Dellenmark-Blom³  Kate Abrahamsson⁴  John Eric Chaplin⁵ 

[1] Department of Medical Psychology, University Medical Center Hamburg-Eppendorf, Martinistraße 52 – W26, 20246, Hamburg, Germany;Department of Pediatric Surgery, Hannover Medical School and Auf der Bult Children’s Hospital, Carl-Neuberg-Straße 1, 30625, Hannover, Germany;Department of Pediatric Surgery, Queen Silvia Children’s Hospital, Sahlgrenska University Hospital, Drottning Silvias Barn O Ungdomsjukh, Rondvägen 10, 41685, Göteborg, Sweden;Department of Pediatric Surgery, Queen Silvia Children’s Hospital, Sahlgrenska University Hospital, Drottning Silvias Barn O Ungdomsjukh, Rondvägen 10, 41685, Göteborg, Sweden;Institute of Clinical Sciences, Department of Pediatrics, Gothenburg University, The Queen Silvia Children’s Hospital, 41686, Gothenburg, Sweden;Institute of Clinical Sciences, Department of Pediatrics, Gothenburg University, The Queen Silvia Children’s Hospital, 41686, Gothenburg, Sweden;

关键词: Esophageal atresia;    Congenital malformation;    Rare disease;    Health-related quality of life;    Parent–child agreement;   

DOI  :  10.1186/s13023-021-01748-x

来源: Springer

【 摘 要 】

BackgroundThe aim was to compare parent and child-reported health-related quality of life (HRQOL) of children born with esophageal atresia (EA) and determine factors that affect the level of parent–child agreement.MethodsWe included 63 parent–child dyads of children born with EA aged 8–18 from Germany and Sweden. The generic PedsQL 4.0™ questionnaire and the condition-specific EA QOL questionnaire were used to assess children’s HRQOL from parents' and children’s perspectives. The PedsQL™ Family Impact Module was used to assess parental HRQOL and Family Functioning.ResultsOn an individual level, intra-class correlation coefficients indicated strong levels of parent–child agreement (.61–.97). At the group level, the analyses showed no significant differences between the responses of parents and children. When a disagreement occurred, parents were more likely to rate generic HRQOL lower than the children (19–35%) and condition-specific HRQOL higher than the children (17–33%). Findings of the binary logistic regression analyzes showed that the child’s age, gender, and country (Germany vs. Sweden) were significant predictors of parent–child agreement in condition-specific HRQOL. We did not identify any significant variables that explain agreement for the generic HRQOL.ConclusionThe parent–child agreement is mostly good, suggesting that parent-reports are a reliable source of information. However, discrepancies may occur and can be explained by the child's age, gender, and country (Sweden vs. Germany). Both perspectives are essential sources for treating EA patients and should not be considered right or wrong. Instead, this information broadens the perspective on pediatric EA patients.

【 授权许可】

CC BY