Results from the validation of the CarCGQoL questionnaire in a sample of caregivers of children with congenital cataract, ROP, and corneal disorders revealed the questionnaire to possess acceptable measurement precision (reliability), but with misfitting items for each condition, individually, and as a pooled sample. After iterative item removal, three different versions of the questionnaire (one for each diagnostic condition) emerged from this validation process. Six items demonstrated misfit; four items (“anger,” “guilt,” “anxiety,” “powerlessness in facing child's disease”) were dissimilar across the three diagnostic groups, and two items (“marriage prospects” and “see after surgery”) demonstrated misfit consistently across the three groups and in the total group (combined sample) of participants. The four items that demonstrated misfit across the three groups were those that addressed the emotional well-being of participants. Given the overlapping item content in the questionnaire that addresses the emotional well-being (albeit not the same aspects such as “depression” and “sleepless nights”), and the focus group discussions we had with caregivers regarding these aspects during the development of this questionnaire previously, we believe that the remainder of the items sufficiently tap into the emotional well-being and thereby the QoL issues of the caregivers of children with ocular conditions. Thus we believe that face validity of the questionnaire is acceptable, thereby providing confidence to future users of the questionnaire.
As noted earlier, two items (Q 19 “marriage prospects” and Q 20 “ability to see after surgery”) demonstrated misfit consistently across all the three diagnostic groups. This finding was unexpected given the origin of items from a cohort of caregivers of children with eye disorders, albeit PCG, during the development of the questionnaire. Items misfit for a variety of reasons such as poor construction and, therefore, risk being poorly understood or may be ambiguously worded. Perhaps this was the reason for poor fit of one of these two items (Q20). Whereas “ability to see after surgery” lacks qualifiers such as see caregiver's face, toys, inanimate objects, faces of familiar people, or any age-appropriate object, these may be considered by users in future studies. Despite these changes, if it continues to demonstrate misfit, then rather than deleting it completely from the questionnaire, it could be analyzed separately such as in a pre-post design study. In case of the other item (Q19), a more likely explanation for the misfit may be related to missing responses to this item (32% of participants) indicating that most of the caregivers never thought of these issues. Given the high proportion of missing responses to this item, we considered it appropriate to delete it. Health-related assessment tools that lack measurement equivalence across population subgroups can result in flawed research and erroneous clinical decisions.
We investigated whether the pooled sample respond to the items of the CarCGQoL questionnaire in the same way as the reference group (caregivers of children with PCG). This is an important prerequisite to determine whether using the CarCGQoL questionnaire is justified in caregivers of congenital cataract, ROP, and corneal disorders and whether their CarCGQoL scores can be interpreted in the same way as in caregivers of children with PCG. The pooled sample responded significantly differently to two items (“powerless in facing child's disease” and “lack of interest to pursue any leisure activity”) out of a total of 20 items (10%), when compared with the reference group. Regardless of the level of their QoL, the pooled sample was more likely to endorse these two items compared to the caregivers of PCG. This would mean that if a caregiver of a child with congenital cataract, ROP, and corneal disorders has high QoL, yet endorses these two items, the questionnaire score could be lower than for a caregiver of a child with PCG with lower QoL. This perhaps suggests that not all of the items in the CarCGQoL questionnaire assess caregiver's QoL equally across those with PCG versus different childhood ocular conditions. Therefore caution should be exercised when comparing the results of the QoL of caregivers of children with PCG and other childhood ocular conditions, or when pooling the data in a single analysis of CarCGQoL questionnaire. Although problems with translation of items in a questionnaire may be one of reasons for an item to be flagged for DIF, we believe that this may not be the case in our study given that none of these items demonstrated DIF by language. Even though the CarCGQoL questionnaire was tested to ensure that the questions are worded properly during the development process, there still remains the possibility that certain groups of caregivers not previously examined may have different interpretations. However, in the present study, all the caregivers were provided with a choice of language among the three available and had the opportunity to ask for clarifications if they were confused. Although we do not have a ready explanation for the occurrence of DIF in the present study, the likelihood of poor wording being the primary cause of the DIF found between the PCG group and other childhood ocular conditions appears remote, and rewording or removal of items is probably unwarranted. Although the deletion of these two DIF-causing items did not appear to have an appreciable impact on the measurement precision (0.84 logits) and targeting (0.62 logits), these two items are still relevant because they tap into social functioning and may be providing little QoL-related information for one cohort but valuable information for another. Replication is clearly needed to support this and all of the findings presented here. Should it be necessary to compare the caregiver's QoL between PCG and other childhood ocular conditions, special attention should be paid to these two items that exhibit DIF.
24,25
The CarCGQoL questionnaire demonstrated acceptable measurement precision (reliability > 0.80), which indicates that it can separate at least three distinct groups of caregivers with perceived QoL, thus supporting its use as a measure.
26 Furthermore, our results showed that the CarCGQoL items form a unidimensional scale within each caregiver group, which confirms that the items measure one underlying construct and that the summation of individual item scores to create a total score is justified in caregivers of children with corneal disorders, ROP, and congenital cataract. Taken together, these results support the validity of the CarCGQoL questionnaire for each diagnostic group tested, thus supporting its generic measurement properties.
Our results showed that, in accordance with the item hierarchy in the original scale among caregivers of children with PCG, items such as “experiencing depression,” “experiencing anxiety,” and “having sleepless nights” were most the difficult and “feeling guilty” and “experiencing irritability” were the easiest to endorse by the participants.
1 The intermediate items showed some, albeit minor, variation in item hierarchy when compared with the PCG sample. As can be seen from
Table 2, targeting of item difficulty to person ability was ideal only for the ROP group (0.39 logits) but was still within the acceptable range (<1.0 logit) for the other two groups (0.61 and 0.74 logits) and for the combined sample (0.62 logits). However,
Figure 2 shows that there were many redundant items, as well as a lack of “hard” items to target the “most able” participants (at the top). This suggests that the questionnaire has relatively better precision to reliably detect differences and changes in caregivers with worse QoL as compared to those with better QoL. The results of the present study regarding the optimal psychometric properties of CarCGQoL questionnaire across different diagnostic groups are perhaps not surprising given that the psychological, financial, and social issues that a caregiver has to face when his/her child is initially diagnosed with an eye disorder are similar, be it PCG, congenital cataract, ROP, or corneal disorders. Nonetheless, as mentioned earlier, psychometric validity in another patient group cannot be assumed and needs to be tested and demonstrated as has been done in the present study.
There are a few limitations in our study. Caregivers of children with ROP were significantly younger compared to those of congenital cataract and corneal disorders and reported comparatively much worse QoL. Given this, the possibility that at least a portion of the variations in the caregiver's QoL associated with age difference may be the result of DIF rather than actual difference in QoL cannot be excluded. The samples analyzed in our study are based on a combination of strategic and convenience sampling procedures. It is therefore difficult to draw general conclusions from the findings of the study to a larger representative sample. However, the variations in diagnostics and the wide range of demographics in combination with rather large sample sizes may contribute information that may be clinically relevant for pediatric ophthalmologists.
It should be acknowledged that, the responsiveness of the questionnaire to intervention has not been assessed as yet. Nevertheless, the results of the present study highlight the importance of better understanding the role of child's ocular condition on caregiver's QoL and suggest areas of future research. For example, the impact of chronic childhood ocular conditions that are not life-threatening versus those that are life-threatening (such as retinoblastoma), on the caregiver's QoL can be attempted in future studies.
Although our sample refers to a very specific population of caregivers of children with congenital cataract, ROP, and corneal disorders, we believe that several challenges that are encountered by this group are also shared with other difficult caregiving scenarios. As such, further research is warranted to replicate these findings among other caregiver populations, for example, in caregivers of children with syndromic ocular conditions, retinoblastoma, and more.
In conclusion, this analysis of the cross-diagnostic validity of the CarCGQoL questionnaire shows that care must be taken when data from different pediatric ocular conditions are pooled given the presence of DIF between the reference group (PCG) and the pooled sample. This suggests that when evaluating the impact of interventions on the caregiver's QoL in a pooled sample, cross-diagnostic DIF must be taken into account. However, it must be emphasized that before any recommendations can be made for modifying the CarCGQoL questionnaire based on DIF, the results found here must be replicated with other pediatric ocular conditions as well. This study suggests that caregivers of children with different pediatric conditions do not define QoL in exactly the same manner, and research involving QoL that uses different pediatric conditions cannot ignore this important issue. The continuing misfit of some items in three different diagnostic conditions resulted in three different versions of the CarCGQoL, but these condition-specific versions are valid and reliable for measuring the QoL of caregivers of children with chronic pediatric ocular conditions. It is an easy instrument to administer and can be completed by the caregiver before the first meeting with the ophthalmologist, which is an advantage when having to deliver intervention in time constrained health care service. This is the first attempt to test the cross-diagnostic validity of the CarCGQoL questionnaire, and we hope that there will be increased research in this area.