This study investigated the parent-child-agreement of HRQOL in a sample of children and adolescents born with EA and the 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, Rohenkohl (13) 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 (8, 28).
Contrary to previous studies' results (13, 29), 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 (1). However, a proportion of child-parent dyads also demonstrated directional differences in children’s generic and condition-specific HRQOL ratings. 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. Parents of children with rare, chronic diseases often experience their child's diagnosis as a traumatic experience that completely changes their previous life and plans (30). Furthermore, they tend to compare their child's health and quality of life with healthy peers because of their loss of the ‘perfect’ child (31). This comparison explains the underrating of the generic HRQOL compared to the perspective of the affected children and adolescents.
Simultaneously, the parents were more likely to score their children’s HRQOL higher than the children when using the condition-specific tool, except the Body Perception domain. A possible explanation relates to the nature of the different measurement levels' questions. A condition-specific instrument is more sensitive to clinical characteristics and raises relevance for the patient group. The EA-QOL© questionnaire was developed using the child's experiences of primary importance and parents’ as complementary importance. Therefore, it might be easier for children to answer those questions (26, 32). The intensity of the experienced limitation and the most recent experiences are essential for assessing the subjective HRQOL (33, 34). Since children and adolescents born with EA experience their chronic health condition directly, they might experience condition-specific aspects of HRQOL more burdensome than generic aspects. Hence, children and adolescents rate their HRQOL lower than their parents.
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 the parent’s perspective and the 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. Except for 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 (5). Our results are consistent with previous research in children with chronic diseases, which found that parents tend to underestimate their child’s HRQOL (5, 8, 13, 14, 35, 36). 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 (1, 26, 32).
On the domain level, our findings showed that the lowest rates of agreement were present in the generic domain Physical Functioning and the condition-specific domain Eating. Both domains can be regarded as observable HRQOL dimensions for the parents that are not related to the child's internal experiences. The level of agreement was found to be highest in the domain Social Functioning using the PedsQL 4.0TM while using the EA-QOL©, the agreement was highest in the domain Health & Wellbeing. These results contrast with previous research, which describes better agreement for observable dimensions (8, 37, 38). These results underline 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 information (age, gender, country) contributed to explaining a significant variation of the parent-child disagreement, but only for the condition-specific EA-QOL© questionnaire and not for the generic HRQOL. Our results showed that parents underestimate children’s HRQOL when the children are younger and overestimate children’s HRQL when the children are older. We assume that children use their peers to talk about personal stress and burden with increasing age. Parents are no longer close to their children's experiences and may interpret less communication as an improvement in coping. The current literature is inconsistent regarding age's effect on the parent-child agreement. Some studies described higher agreement in younger children supporting the hypothesis that increased independence during puberty may limit the exchange between parents and children (38, 39). In contrast, other studies found higher levels of agreement for older children attributed to growing cognitive and communication skills (40, 41). However, some studies did not find a significant influence of child age on the level of parent-child agreement (37). Nevertheless, only a few studies consider this variable in statistical analysis (9, 42).
Clinical and parental characteristics did not significantly affect the parent-child agreement in EA patients and their parents. Other studies reported that the level of agreement between parents and children seemed more strongly associated with familial and social factors (13, 28, 43). The residence country explained the underestimating of parents in our study using the condition-specific tool. It is difficult to identify a definite explanation for these results. However, there might be different norms or traditions of how parents and children in different countries communicate about their health condition and its consequences in daily life (44).
Regarding the child’s gender, we showed that being a boy was associated with a higher relative probability of parents’ underestimating children’s HRQOL. Being a girl increases the probability of parents’ overestimating children's HRQOL. In contrast to our results, other studies reported that the child’s gender did not significantly affect the parent-child agreement (45-47). Waters, Stewart-Brown (48) reported that parents and a female child's dyads showed lower agreement than parent-boy dyads. However, this study only included adolescents. Gender and age are rarely examined in studies and need to be investigated in more detail.
Limitations
Some limitations should be taken into account when interpreting the present study results. Concerning the sample, it should first be mentioned that the parents’ perspective mostly consisted of mothers’ reports due to fathers' low response rate. Even though this is not uncommon in pediatric health care studies and clinical practice (9), the results are biased towards the mothers’ perspective. They should be replicated in future studies using larger fathers' samples to determine whether there are differences between father and mother reports. Although this sample size is relatively large for a rare disease study such as EA, the small sample size of 63 parent-child-dyads and the different sample composition in Germany and Sweden does not allow us to draw meaningful conclusions. The missing data for single domains must be considered when interpreting the results. Missing data resulted from single items that were not answered by individuals. Since both parents and children's data had to be available in full to compare the perspectives, dyads had to be excluded as soon as the domains could not be calculated for both. This small sample size should also be considered when interpreting the 95% CI.
Moreover, our study sample seems fairly representative regarding gestational age at birth, primary esophageal repair, and child gender. However, the prevalence of associated anomalies seems slightly higher than in previous reports, especially cardio-vascular anomalies (49).
Another limitation concerns the methods of data collection. Since families have filled out the questionnaires at home, a parental influence of the children’s answers cannot be excluded even though parents have explicitly been asked not to influence their children’s answers. This study's results can be used as an indication of future studies investigating parent-child agreement in pediatric patients with rare malformations such as EA.
Conclusion
Of importance to clinical practice and future research, the parent-child-agreement when rating EA children’s HRQOL is mostly good, suggesting that parent reports are a reliable source of information. However, discrepancies may occur, which may differ in the direction in the child-report depending on generic or condition-specific measurement level. The child's age, gender, and country origin (PR<CR vs. agreement) can explain the parent-child-agreement using condition-specific HRQOL measurements. These variables need to be investigated in larger samples and need to be considered when interpreting HRQOL measures of EA patients and their parents. Nevertheless, the various 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.
Key messages
- Agreement between child and parent reports when rating EA children’s HRQOL is mainly good, suggesting that parent-reports are a reliable source of information.
- In EA children, the directional discrepancy in parent-report vs. child-report shows parents' tendency to underrate their children’s generic HRQOL and overrate the condition-specific HRQOL.
- In condition-specific HRQOL assessment of EA children, disagreement between child- and parent-report may be explained by the child’s age, gender, and country origin (Sweden vs. Germany).