This study investigated the parent-child-agreement of HRQOL in a sample of children and adolescents born with EA and determinants of discrepancies. Inconsistent with previous findings from the literature, which advocate moderate levels of agreement in pediatric HRQOL assessment, we have found strong levels of agreement between child- and parent-reported children’s HRQOL on the individual level. While Quitmann et al. (2016) also report at least moderate to good ICC levels for children and adolescents with short stature and their parents, other studies reported only moderate levels of parent-child agreement in chronic diseases (Eiser & Varni, 2013; Silva, Crespo, Carona, Bullinger, & Canvarro, 2015).
Contrary to the results from previous studies (Quitmann et al., 2016; Silva et al., 2013), we found no differences in the agreement between generic and condition-specific instruments. According to our results, the agreement was good between children and parents (Dellenmark-Blom et al., 2018). However, a proportion of child-parent dyads also demonstrated directional differences in the rating children’s generic and condition-specific HRQOL. Interestingly, the direction of the differences between parent- and child-reported children’s HRQOL differed depending on the instrument used. Parents tended to underrate children’s HRQOL using generic HRQOL measures. At the same time, the parents were more likely to score their children’s HRQOL higher than the children when using the condition-specific tool, with the exception of the domain Body Perception. A possible explanation relates to the nature of the questions of the different measurement levels. A condition-specific instrument is more sensitive to clinical characteristics and raises issues of relevance for the patient group. The EA-QOL questionnaire was developed using the child experiences of primary importance, and parents’ as complementary importance. Therefore, it might be easier for children to answer those questions (Dellenmark-Blom et al., 2017; Dellenmark-Blom et al., 2016).
Approximately half of the dyads showed an agreement – defined as differences of equal or less than half of the standard deviation of the score - between parent’s perspective and children’s perspective. When disagreement occurred, it was likely to be in the direction of parents underrating children’s HRQOL using the generic HRQOL measurement. In all domains, with exception of the domain Social Functioning, the underrating was present in approximately one-third of parents. The same pattern was found in other studies of children with chronic health conditions (Silva, Crespo, Carona, Bullinger, & Canavarro, 2015). This is consistent with previous research in children with chronic diseases, which found that parents tend to underestimate their child’s HRQOL (Eiser & Varni, 2013; Levi & Drotar, 1999; Quitmann et al., 2016; Rohenkohl et al., 2015; Sheffler et al., 2009; Silva, Crespo, Carona, Bullinger, & Canavarro, 2015). Inconsistent with these findings, the direction of disagreement we found in our sample for the condition-specific HRQOL measurement was opposed. One-third of parents overrated their children’s HRQOL, especially in the domains Eating, Social Relationships, and the Total Score. Here again, it might be that the questionnaire was more sensitive to the children’s perspective than the parents’ since it was developed primarily according to the children’s experiences (Dellenmark-Blom et al., 2017; Dellenmark-Blom et al., 2016; Dellenmark-Blom et al., 2018).
On the domain level, our findings showed that the lowest rates of agreement were present in the generic domain Physical Functioning as well as in the condition-specific domain Eating. Both domains can be regarded as observable HRQOL dimensions for the parents that are not related to the internal experiences of the child. Using the PedsQL, the level of agreement was found to be highest in the domain Social Functioning, while using the EA-QOL, agreement was highest in the domain Health & Wellbeing.” Thus, these results contrast with previous research, which describes better agreement for observable dimensions (Eiser & Varni, 2013; Patel, Lai, Goldfield, Sananes, & Longmuir, 2017; Rajmil, Rodriguez López, López-Aguilà, & Alonso, 2013). This underlines the importance of capturing the child-report using the EA-QOL questionnaire in clinical practice when monitoring and providing supportive interventions to the child’s HRQOL.
In our analyses, sociodemographic (age, gender) and clinical variables (EA severity level) did not contribute to explaining a significant amount of variation of the extent of parent-child discrepancies. The level of agreement between parents and children instead seemed more strongly associated with familial and social factors (Quitmann et al., 2016; Silva, Crespo, Carona, Bullinger, & Canvarro, 2015; Van Roy, Groholt, Heyerdahl, & Clench-Aas, 2010). The current literature reported diverse findings on variables explaining parent-child-agreement. While some studies found higher levels of agreement for older children and explaining this by growing cognitive and communication skills (Annett, Bender, DuHamel, & Lapidus, 2003; Peetsold, Heij, Deurloo, & Gemke, 2010), other studies described higher agreement in younger children supporting the hypothesis that increased independence during puberty may limit the exchange between parents and children (April, Feldman, Platt, & Duffy, 2006; Rajmil et al., 2013). However, there were also studies, which did not find a significant influence of child age on the level of parent-child agreement (Patel et al., 2017). So far, there are no consistent results on the effect of child gender on the parent-child agreement, yet there are only a few studies considering this variable in statistical analysis (Eiser & Morse, 2001; Upton et al., 2008). The country of residence only explained the parent-child-agreement in our study when using the generic tool. It is difficult to identify a definite explanation for these results, but there might be different norms or traditions of how parents and children in different countries communicate about the child’s health condition and its consequences in daily life (Matza et al., 2013). No other variables in the model contributed significantly to the extent of parent-child-agreement on generic HRQOL. The extent of disagreement of the condition-specific measurement was explained by the parent-reported parental HRQOL. The lower the parental HRQOL, the higher the level of parent-child discrepancies. In other studies parents with higher parental stress, especially depressive symptomatic reported significantly more limitations of their children than parents without these stressors (Kobayashi & Kamibeppu, 2011). This correlation has been more strongly demonstrated for mothers than for fathers (Davis, Davies, Waters, & Priest, 2008), and most parent-reports in this study were maternal perspective. However, Eiser and Varni (2013) add that the majority of research has taken into account primarily the maternal perspective, meaning that ratings of fathers or differences between mothers and fathers are only permitted to a limited extent. Indeed, it would be relevant for everyday clinical practice whether there are systematic differences in the assessment of HRQOL of children between mothers and fathers, which they have to be considered by the clinicians.