Current Pediatric Research

Abstract

Background: Child involvement in health care decision-making is essential during health planning. Additionally, parents’ perceptions of their children’s Health-Related Quality of Life (HRQOL) could influence healthcare utilization in regards to children with Cerebral Palsy (CP) and their families.

Objectives: Our goal was to assess the level of agreement between children with CP and their parent’s perception of HRQOL by using the PedsQL™ 3.0 instrument in Arabic.

Materials and Methods: a cross-sectional study involving a convenient sample of 29 children with CP (mean age of 10.3 ± 1.5 years old) and their parents/caregivers filled the electronic version of the PedsQL™ 3.0 Cerebral Palsy Module in Arabic. The data was analysed via Pearson’s Correlation Co-efficient (PCC) and Intraclass Correlations Coefficient (ICC).

Results: We found a strong to moderate positive correlation between child self-reports and parent proxy-reports in the Daily Activity (r=0.58, p<0.001); Movement and Balance (r=0.61, p<0.001); and Eating Activities (r=0.58 p<0.001). A moderately positive correlation was found in the Speech and Communication scale (r=0.48, p<0.001) of PedsQL™ 3.0. There was no significant correlation in the School Activity, Fatigue, and Pain and Hurt scales. The parents/caregivers significantly underestimated their children’s QOL in the School Activity, Pain and Hurt, and Movement and Balance scales.

Conclusion: An acceptable level of agreement was found in certain PedsQL™ 3.0 scales. However, we found a tendency for parents/caregivers to underestimate their child’s QOL. Future studies should include a larger sample size and validate the Arabic translation of the PedsQL™ 3.0.

Keywords

Quality of life, Cerebral palsy, Saudi, Children, Health-related quality of life, PedsQLP™, HRQOL.

Introduction

Ronsenbaum et al. defines Cerebral Palsy (CP) as ‘a group of permanent disorders of the development of movement and posture, causing activity limitation, that are attributed to nonprogressive disturbances that occurred in the developing fetal or infant brain’ [1]. CP is the second most common disability among children in the Kingdom of Saudi Arabia (KSA), with a prevalence rate of 23.4 per 10,000 children and 14% of CP cases are located in the central province [2]. Children with CP experience a wide range of motor impairments, which places heavy demands on health, education, and social services. The condition may be accompanied by additional impairments that affect people with CP’s daily life and participation in society, such as neuroimpairments, which include spasticity, muscle weakness, and limited range of motion that affects gross and fine motor function [3], and cognitive and sensory impairments, such as a learning disability, epilepsy, and speech and visual impairments [2,3]. This condition could adversely affect the Health-Related Quality of Life (HRQOL) of children with CP and their families. HRQOL is defined as “how well a person functions in their life and his or her perceived wellbeing in physical, mental, and social domains of health” [4].

The involvement of children in health care decision-making during health planning demonstrates respect for their capabilities and promotes a sense of control and cooperation with health professionals [5]. Children can reliably report their HRQOL when provided with a questionnaire appropriate to their age and cognitive function [6]. Varni et al. demonstrated that children as young as 5 years old could reliably and validly self-report their HRQOL when given an age-appropriate instrument [7]. Parents also have a fundamental role in the decision-making process for their child’s health care. Their role enhances physician-parent-child communication and reflects positively on patient satisfaction and compliance [5,6]. Children who are too young or have intellectual disabilities, such as children with CP, may not reliably self-report their HRQOL. Hence, to optimally assess outcomes in such cases, parent proxy-report instruments should be used as either a primary or secondary outcome measure [8,9]. Additionally, parents’ perceptions of their child’s HRQOL could influence health care utilization [5,7]. Therefore, an HRQOL instrument for children with CP should measure the perspectives of both children and parents using the same scales with parallel items to provide comparable results between self-reports and proxyreports [7].

Recent studies [10,11] show a high agreement between child self-reports and parent proxy-reports in the perception of HRQOL among children with Attention Deficit Hyperactivity Disorder and epilepsy. However, other studies in the literature report that parents of children with CP tended to underestimate their child’s Quality of Life (QOL), since the quality of life was rated significantly higher by the children than by their parents [6,12,13]. In addition, culture and society could influence parents’ perspectives on quality of life [14].

Therefore, it is important to assess the level of agreement between children with CP and their parents according to their culture and the country they live in. The PedsQL™ 4.0 Generic Core Scales is a valid and reliable self-report instrument to measure HRQOL outcomes in children and adolescents from 2 to 18 years old [15], and it includes both a child self-report and a parent proxy-report [7]. An earlier version, the PedsQL™ 3.0 Cerebral Palsy Module, was designed for children and adolescents with CP [8,16,17] and translated into different languages, including Arabic, according to the standardized procedures of the MAPI Research Trust [18–22].

However, to the best of our knowledge, no studies have tested the agreement between children with CP and their parents using the PedsQL 3.0 Cerebral Palsy Module. With the growing importance of relying on both child and parents' proxy to utilize health care services. Therefore, the aim of the study was to assess the level of agreement between children with CP and their parent’s perception on their children’s HRQOL using the Arabic PedsQL™ 3.0 Cerebral Palsy Module.

Materials and Methods

Participants

The study has a cross-sectional design utilizing non-probability sampling techniques. A convenient sample of 29 children and their parents participated. Participants included children with CP ages 8 to 12 and one of their parents/caregivers each. The children’s level on the Gross Motor Function Classification System (GMFCS) was disregarded. Children who were diagnosed with other neuromuscular disorders, intellectual disabilities, visual or hearing impairments, or behavioural disorders were excluded from this study.

PedsQL™ 3.0 cerebral palsy module

PedsQL inventory is a valid and reliable outcome measure to assess HRQOL of many health conditions. It provides both child self-report and parent proxy-report [16,17]. The MAPI Research Trust allowed the authors to use the translated Arabic version for both the children’s and parents’ versions of the PedsQL™ 3.0 Cerebral Palsy Module. The child self-report is identical to the parent proxy-report, except for the identifier “you”, which was changed to “your child”. Both reports are comprised of 35 items divided into seven scales: nine items in Daily Activities, four items in School Activities, five items in Movement and Balance, four items in Pain and Hurt, four items in Fatigue, five items in Eating Activities, and four items in Speech and Communication [16,17]. The participants rate the items on a five-point Likert scale from 0 (Never) to 4 (Almost always); thus, higher scores indicate lower problems in HRQOL. The scoring method consists of two steps: i) the score items are reverse scored and linearly transformed into a 0-100 scale as follows: 0=100, 1=75, 2=50, 3=25, 4=0; ii) to account for differences in sample sizes, if more than 50% of the items in the scale are missing, the Scale Scores should not be computed. There is no total score. Instead, the mean score can be calculated through the sum of the items divided by the number of items answered [16,17].

Procedures

The participants were recruited from four different paediatric rehabilitation outpatient clinics in Riyadh: the Prince Sultan bin Abdulaziz Humanitarian City (SBAHC), the Disabled Children’s Association (DCA), the Stand-Out rehab centre, and the Bedayah Specialist Center for Physical Therapy. The data collection process was based on a face-to-face interview with the parents/caregivers and their children. Children and parents were separated from each other and asked to fill out the Arabic version of the PedsQL™ 3.0 Cerebral palsy module on two iPads. Two researchers were available to provide further explanation to ensure that all parties reached full understanding of the process, in order to obtain more in-depth information on the sample. Before the data collection process, certain general questions such as “what do you like to do at school/home” were asked to establish a good rapport with the children in order to increase their comfort during interactions and their level of engagement [23]. Additionally, this was a helpful way to determine whether the cognitive level of the children was appropriate enough and if they understood the task before filling out the questionnaire. Subsequently, demographic data were collected from both the children and their parents/ caregivers by using a simple questionnaire designed by the researchers to collect data on sex, age, social status, parents’ level of education, the child’s current school grade, and how many hours a day the parents spent with their children on average.

Ethical consideration

The study was approved by the IRB committee of the Princess Nourah bint Abdulrahman University (PNU) (IRB log number 18-0252). We obtained informed consent from parents/ caregivers and informed assent from the children. All data collected on the participants is confidential.

Data Analysis

The statistical analysis was performed using the SPSS v.17.0 (SPSS Inc., Chicago, IL, USA) package for MacOS. Sample characteristics were averaged using mean and standard deviation for quantitative variables and percentages for qualitative variables. Internal consistency of the Arabic version of PedsQL™ 3.0 Cerebral Palsy Module (ages 8–12) was assessed using Cronbach’s alpha coefficient for both child selfreport and parents proxy-report. The correlation between the child self-report and parent proxy-report was analysed via Pearson’s correlation coefficient. The level of agreement between child self-report and parent proxy-report was analysed via the Intraclass Correlation Coefficient (ICC) as follows: ≤0.4=poor; 0.41-0.60=moderate; 0.61-0.80=good; and 0.81-1=excellent [24]. Differences in parent and child perception of QOL were assessed using the independent samples t-test. The significance level was set to ≤ 0.05.

Results

Twenty-nine children with CP (mean age 10.3 ± 1.5) and twenty-nine parents/caregivers (mean age of 37.7 ± 9.46) participated in the study. 55% of participants were male, and 69% of proxy-reports were from mothers. The sample data are shown in Table 1.

Table 1. Sociodemographic characteristics of the sample.

A strongly positive correlation was found between parents’ and children’s reports in three scales: Daily activities (r=0.58, p<0.001); Movement and Balance (r=0.61, p<0.001); and Eating activities (r=0.58 p<0.001). A moderately positive correlation was found in the Speech and Communication scale (r=0.48, p<0.001). No association was found between the reports in three scales: School activities; Pain and Hurt; and Fatigue. Results also show a good level of agreement between PedsQL™ 3.0 parent proxy-reports and child self-reports in three scales: Eating Activity (ICC=0.74); Daily Activity (ICC=0.72); Speech and Communication (ICC=0.64). A moderate agreement was found in the Movement and Balance scale (ICC=0.49). However, poor agreement was found in the Fatigue, School Activity, and Pain and Hurt scales (ICC=0.41, 0.15, 0.08 respectively). Data on correlation and agreement can be found in Table 2.

The results of the t-test when comparing the mean and

Table 2. Correlation between the PedsQL™ 3.0 CP parent proxy-reports and child self-reports.

Discussion

The study assessed the level of agreement between children

Table 3. Mean and Standard Error (SE) comparison of the PedsQL™ 3.0 CP parent proxy-reports and child self-reports.

The results indicate a moderate to good agreement between children self-reports and parent proxy-reports of the Arabic version of PedsQL™ 3.0 Cerebral Palsy Module in four scales: Daily activities; eating activities; movement and balance; and speech and communication. Similarly, Majnemer et al. demonstrated that the level of agreement between parents and children with CP varies depending on the scale [25]. The highest level of agreement reported in their study was with respect to physical health and well-being and the lowest agreement, to School Activities. The agreement between the child and the parent could be attributed to the proxy-report rater relationship; mothers usually are the primary caregivers for their children and thus they are more aware of their children’s HRQOL [26,27]. In this study, mothers represented 69% of caregivers and 80% of mothers spent about 12-24 hrs a day with their children. Moreover, Daily Activities, Movement and Balance, and Eating Activities can be more easily observed and assessed by parents than other scales [27,28]. Eiser et al. similarly indicate that the physical functions that could be observed, therefore, showing higher agreement d is not observed [27]. On the other hand, three scales demonstrated poor agreement: School Activity, Pain and Hurt, and Fatigue. Additionally, parents reported significantly lower QOL scores compared to their children in these scales. This result was consistent with several studies that indicate that parents tend to underestimate their children’s QOL [6,12,13]. White-Koning et al. demonstrated that parents OD children with CP tend to underestimate their children’s performance in most of the health-related quality of life [12].

Several factors could justify the discrepancy between parent proxy-reports and children self-reports in specific scales. Since the pain and hurt and the fatigue scales are subjective measurements, parents may be less accurate when assessing this problem [28,29]. For example, children may interpret that fatigue is a result of a certain activity or exertion, or that it may be linked to their mood. On the other hand, the parents/ caregiver may interpret fatigue as a medical condition [29,30]. For the pain and hurt scale, children may think of one specific event while their parents/caregivers consider several events when answering the questions. Moreover, this result could be due to children’s limited ability to understand the concept of pain when compared to their parents. In this study, children were not consistent in responding to questions in the Pain and Hurt and Fatigue scales similarly to their parents, which raises the issue of reliability of those scales in the Arabic version of PedsQL™ 3.0. In the Arabic translation, there was an overlap between the concept of pain and fatigue with most participants, and interpretation of these words may vary according to each participant.

The School Activity scale achieved poor agreement, similar to Varni et al. who obtained a low level of agreement in the school functioning scale on PedsQL™ 4.0 Generic Core Scales [8]. Evidently, some parents are unavailable when their children are performing certain activities at school. Thus, the parent/caregiver might be unaware of their child’s capability at school [29,30]. The results from this pilot study show that parent proxy-reports achieved acceptable to good internal consistency in all scales of the PedsQL™ 3.0 cerebral palsy module in arabic. Nevertheless, child self-reports demonstrated acceptable internal consistency only in the daily activities and eating activities scales. As for the level of agreement, results indicate a good agreement between parents and children in the scales that demonstrated good internal consistency (Daily activity and eating activity). Additionally, those scales were observable functions therefore, contributing the good agreement [28]. The scales that showed poor agreement were those reached unacceptable internal consistency. This may influence the level of agreement among them, suggesting that a higher ICC may result in a higher level of agreement.

The Arabic version of PedsQL™ 3.0 Cerebral Palsy Module should be culturally adapted to accommodate the needs of the population. In this case, the School Activities scale has two questions about keyboards and mouse usage, which are rarer in schools nowadays. Therefore, replacing them with other items that might be more common in their daily lives, such as tablets, video game consoles, and smartphones is recommended. Moreover, in the Eating Activities scale, the type of cuttingwhether by knife or by hand-was not specified. This question need to be modified since in Saudi Arabia, it is culturally acceptable to use one’s hands while eating. Thus, although translated, the PedsQL™ 3.0 is not yet culturally adapted for the Saudi population. This study has several limitations; the first of which is that the level of GMFCS was not assessed, since the functional level of the children may affect the level of agreement. Therefore, further studies should evaluate the functional level of participants. Furthermore, it is recommended that future studies assess the psychometric characteristics of the Arabic version of the PedsQL™ 3.0 Cerebral Palsy Module on a larger sample size, in order to gather enough data to culturally adapt the instrument for the Saudi population.

Although the Arabic version of PedsQL™ 3.0 shows promise, similar studies with larger sample sizes should be conducted to assess the properties of the instrument within Saudi culture. Additionally, further studies are warranted to analyse the psychometric properties of the Arabic version of PedsQL™ 3.0 on a larger scale. For future studies, it is recommended to address the agreement on HRQOL between parents/caregivers and their children in a large sample size with different age groups and disability levels. In addition, further investigation of the factors that may influence this type of agreement is recommended.

Conclusion

The study demonstrates a good agreement between parent proxy-reports and child self-reports across most of the PedsQL™ 3.0 scales. However, other scales show discrepancies. In their reports, parents estimated that their children had a lower level of QOL than the children’s selfreports. This indicates the importance of relying on child selfreports to assess their involvement and access to health care.

Acknowledgment

This research was funded by the Deanship of Scientific Research at Princess Nourah bint Abdulrahman University through the Fast-Track Research Funding Programme.

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