In this study of children treated for non-syndromic CS, we asked patients and their parents to estimate HRQoL using the PedsQL 4.0 Generic Core Scales as a reliable and validated HRQoL-measurement tool specifically constructed for children in pediatric care. Measuring HRQoL with a patient-reported instrument allows access to information directly from the perspective of the patient, which remains a rare but coveted phenomenon in both research and clinical settings. There are few studies examining the subjective perception of the HRQoL of patients treated for non-syndromic CS, with only one study focusing on children treated for CS and reporting results indicating a risk of low HRQoL.24 In another study of untreated SS patients, there was a tendency toward a low score in relation to positive emotions.25
This study, which included a cohort of 73 patients treated for non-syndromic CS, revealed a generally high HRQoL, with no significant differences identified between those treated for CS and norms or between those treated for SS and MS. However, reports of psychosocial and school functioning were lower in the SS group, although no differences in functioning were found between the SS and MS groups according to the estimated HRQoL. Furthermore, the surgical method used for treatment (i.e., spring-assisted surgery and pi-plasty) in the SS group was unrelated to better or worse HRQoL outcomes.
Numerous studies have focused on assessing neuropsychological and cognitive functioning in order to reach a consensus regarding developmental impacts related to CS diagnosis and surgical treatment.6–9, 11-20,22 Previous studies indicate that non-syndromic CS patients are generally expected to exhibit average cognitive development.11,13,15,17,29 The cohort of patients in the present study showed average performance in terms of IQ and ABAS, which were extracted as background data from previous studies.13,26 Notably, we found that associations between HRQoL, IQ, and ABAS were significant, with moderate correlations. This is an important aspect to consider when measuring HRQoL, given that cognitive and adaptive abilities can affect HRQoL outcomes. Therefore, it is crucial to control for these variables in order to measure the intended phenomenon. In a clinical setting, HRQoL could be used to screen patients in need of further psychological assessment.30
Patient-reported outcomes can offer information regarding patient perspective through self- or proxy reports. In this study, we used both self- and proxy reports to assess HRQoL. Interestingly, there were differences in how children and parents estimated HRQoL, with parents inclined to report higher HRQoL than the children. Previous studies frequently used proxy reports, where parents were asked to assess different aspects of developmental questions regarding their child.10,31−34 The results of the present study indicate that it is critical to also use self-reports before drawing conclusions about patient status.
The primary strength of this study is its methodological approach using a validated and reliable measurement, evaluated in a larger group of Swedish schoolchildren, to assess HRQoL. Another methodological strength is the advantages of controlling for confounders through the use of comprehensive amounts of background data. Additionally, the high response rate (80.2%) and the attrition analysis minimized the risk of selection bias. However, there are also limitations, as a larger study group would have increased the probability of the assumptions from the results.