|Year : 2022 | Volume
| Issue : 1 | Page : 16-23
Health-related quality of life in children with juvenile idiopathic arthritis: A developing country perspective
Madhumita Nandi1, Abhisek Majhi2, Moumita Samanta2, Tanmoy Sar2, Sumantra Sarkar3, Tapas Sabui4
1 Department of Pediatrics, In Charge Pediatric Rheumatology Clinic, North Bengal Medical College, Darjeeling, India
2 Department of Pediatrics, NRS Medical College, Kolkata, India
3 Department of Pediatrics, Diamond Harbour Medical College, Diamond Harbour, West Bengal, India
4 Department of Pediatrics, RG Kar Medical College, Kolkata, India
|Date of Submission||12-Sep-2020|
|Date of Acceptance||12-Apr-2021|
|Date of Web Publication||25-Feb-2022|
Dr. Madhumita Nandi
6/6, Naren Sarkar Road, Barisha, Kolkata - 700 008, West Bengal
Source of Support: None, Conflict of Interest: None
Objective: To assess the Health-Related Quality of Life (HRQoL) in domains of physical, emotional, social, and school functioning in children suffering from juvenile idiopathic arthritis (JIA).
Materials and Methods: This hospital-based prospective observational study was undertaken in children from 2 to 12 years of age attending the Rheumatology clinic of a tertiary care teaching institute of Eastern India with a diagnosis of JIA for at least 6 months. Pediatric QoL inventory 4.0 generic core scale was used after obtaining the necessary permission.
Results: Out of a total of 92 patients of JIA, 85 (male/female: 39/46) were taken up for the study polyarticular JIA was the commonest variety (54/85, 63.5%) followed by systemic JIA (sJIA) (25/85, 29.4%) and oligoarticular JIA (6/85, 7.0%). Male gender (P = 0.059, 0.000), better BMI (P = 0.004, 0.001), better disease control (P = 0.00, 0.00), sJIA subcategory (P = 0.002, 0.000), and longer duration of disease (P = 0.015, 0.000) correlated with better QoL in both child and parent scores whereas better socioeconomic status fared better only in parent scores (P = 0.017) and educational status of parents in child scores (child P = 0.000), Child and parent score were comparable across all domains and also overall scores.
Conclusion: This study brings out certain important aspects of QoL in children suffering from JIA which may have some significant impact on disease control per se. Larger community-based multicentric studies would help us in delineating further this aspect of the disease and formulating guidelines for improving QoL in such children.
Keywords: Children, health-related quality of life, India, juvenile idiopathic arthritis
|How to cite this article:|
Nandi M, Majhi A, Samanta M, Sar T, Sarkar S, Sabui T. Health-related quality of life in children with juvenile idiopathic arthritis: A developing country perspective. Indian J Rheumatol 2022;17:16-23
|How to cite this URL:|
Nandi M, Majhi A, Samanta M, Sar T, Sarkar S, Sabui T. Health-related quality of life in children with juvenile idiopathic arthritis: A developing country perspective. Indian J Rheumatol [serial online] 2022 [cited 2022 May 16];17:16-23. Available from: https://www.indianjrheumatol.com/text.asp?2022/17/1/16/338448
| Introduction|| |
Contrary to the definition of health by the World Health Organization as a 'state of complete physical, mental and social well-being,” day-to-day medical practices and clinical studies mostly focus on the analysis and relief of somatic symptoms, often ignoring the mental and social dimensions of health. However, over the last few decades, the term Health-Related Quality of Life (HRQoL) is gradually coming up to draw attention to the social and mental aspects of chronic diseases and is gradually being included in the clinical study protocols to assess treatment outcome of those chronic diseases where we need to focus more on long-term morbidities and disabilities in addition to short-term outcomes.,,,
Although a few studies have been published on HRQoL in some chronic pediatric disorders like thalassemia, asthma, nephrotic syndrome, and cerebral palsy,,,,, there is hardly any on childhood chronic rheumatological disorders, especially from the less developed parts of the world like India, where the long-term management of these patients may not always be optimum because of many financial and resource constraints. The magnitude of the disease burden can be gauged from the fact that an estimated 1.3 million children with juvenile idiopathic arthritis (JIA) are in India alone and the worldwide prevalence of JIA varies from 0.7 to 1.0/1000 children., Chronic joint and other systemic symptoms with not so uncommon resultant disabilities, diagnostic, complication, and management-related frequent stays in hospital, consequent prolonged school absenteeism, and therapeutic agent-related adverse effects have every potential to have a negative impact on the overall QoL of these children, and their parents and families. This study is an effort to assess the QoL of such children and evaluate the effect of multiple diseases and patient variables on different domains of QoL such as physical, emotional, social, school functioning, and psychosocial.
| Materials and Methods|| |
The primary objective of this study was to analyze the HRQoL in physical, emotional, social, school functioning, and psychosocial domains. The secondary objective was to find out the impact of variables such as gender, disease type, socioeconomic status, patient and family characteristics, duration and activity of disease, and treatment modalities adopted on the QoL of these children in each of the specified domains.
Study design and patient selection
This hospital-based prospective observational study was undertaken in children from 2 to 12 years of age attending either Pediatric Rheumatology clinic or admitted indoors of a tertiary care teaching institute of Eastern India with a diagnosis of JIA for at least 6 months. Requisite prior approval from the Institution Ethics Committee was taken IEC/NMC -6552 dated 26.12.2016. Furthermore, written informed consent was taken from the parents/guardians and assent was taken in children above 7 years. Patients with other co-morbid medical or behavioral conditions, those who had defaulted on any drug for more than 6 weeks and those who could not understand Bengali/Hindi/English or those who refused to participate were excluded from the study.
Study technique and tool
After filling up a pretested study pro forma containing all the relevant demographic, social, treatment, and disease activity related background information Pediatric QoL Inventory (Peds QL 4.0) generic core scale was used after obtaining necessary permission from Mapi Research Trust (Marie Sidonie EDIEUX/[email protected]) as a study tool in this non funded research study. The Kuppuswamy's revised (2012) socioeconomic status scale was used for socioeconomic status classification. For recording the educational status, the educational status of the parent with the higher degree was noted in case of discrepancies between the academic degrees of parents.
The PedsQL™ 4.0 is the only empirically validated generic pediatric HRQoL measurement instrument to span this broad age range for child self-report and parent proxy-report while maintaining item and scale construct consistency, feasibility, reliability, and validity., Items on the PedsQL 4.0 have minimal missing responses, suggesting that children and parents are willing and able to provide good-quality data regarding the child's HRQoL. The self-report and proxy-report internal consistency reliabilities generally exceeded the recommended minimum alpha coefficient standard of. 70 for group comparisons.,
The PedsQL™ 4.0 Generic Core scales have 23 items based on physical, emotional, social, and school functioning domains. There are separate questionnaires for children and parent proxy reports for those between 5 and 12 years and only parent proxy report questionnaires for children from 2 to 4 years with 21 items. The items are rated on a 5-point ordinal scale to indicate how much the child has problems with various areas of functioning, ranging from 0 (never) to 4 (almost always) over the last 1 month. For younger children (5–7 years), the ordinal scale is reworded and simplified to a 3-point scale: 0 (not at all a problem), 2 (sometimes a problem), and 4 (a lot of a problem). The items were reverse-scored and linearly transformed to a 0–100 scale as: 0 = 100, 1 = 75, 2 = 50, 3 = 25, 4 = 0. Hence, a higher score after this computation meant a better QoL. If more than 50% of the items in the scale were missing, the scale scores of that particular were not computed. If 50% or more items are completed then the mean of the completed items in that scale was imputed. The mean score was computed as the sum of the items over the number of items answered. Four subscales, including physical functioning (8 items), emotional functioning (5 items), social functioning (5 items), and school functioning (5 items), contribute to 3 summary scores: Total scale score (all subscales), physical health summary score (physical functioning scale only), and psychosocial health summary (emotional, social, and school functioning scales combined).
Data were compiled in worksheet of Microsoft Excel and results were plotted by bar diagram where feasible using Windows 10, standard version. Data were analyzed using SPSS 24.0 and GraphPad Prism version 5 (IBM SPSS Statistics for Windows, Version 24.0. Armonk, NY: IBM Corp GraphPad Prism version 5.01, CA, USA). Data were summarized as mean and standard deviation for numerical variables and count and percentages for categorical variables. Two-sample t-tests for a difference in mean involved independent samples or unpaired samples. Paired t-tests were a form of blocking and had greater power than unpaired tests. One-way analysis of variance (one-way ANOVA) was a technique used to compare means of three or more samples for numerical data (using the F distribution). A Chi-squared test was any statistical hypothesis test, wherein the sampling distribution of the test statistic is a Chi-squared distribution when the null hypothesis is true. Without other qualifications, “Chi-squared test” often is used as short for Pearson's Chi-squared test. Unpaired proportions were compared by Chi-square test or Fischer's exact test, as appropriate. P ≤ 0.05 was considered statistically significant.
| Results|| |
Out of a total of 130 children with rheumatological complaints attending the study institute over a 6 months period, 92 were patients of JIA. Eighty-five (39 males and 46 females) were finally taken up for the study and the data analyzed after excluding the rest based on various criteria as stated above [Figure 1]. Polyarticular JIA (pJIA) was the commonest variety (54/85, 63.5%) followed by systemic JIA (sJIA) (25/85, 29.4%) and oligoarticular JIA (oJIA) (6/85, 7.0%). There were no psoriatic and enthesitis-related arthritis in this series. Among oligoarticular, all were persistent oligoarticular. [Table 1] gives the overall background characteristics of the 3 subtypes of JIA. Of the polyarticular, 3 patients were rheumatoid factor positive. There was no statistically significant difference in the patient characteristics of the subgroups except gender distribution. Female patients were more in number in all individual subgroups as well in overall incidence. Other demographic, epidemiological, anthropometric, and disease activity-related characteristics of the three subgroups were comparable.
|Table 1: Background demographic and clinical information on each category of juvenile idiopathis arthritis patients|
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On gender-wise comparison of HRQoL of the study population, male children, in general, had significantly better scores in all individual domains and also in psychosocial summary scores and global scores on both self and parent assessment scales, more significantly so on most of the parent assessment scales [Figure 2]. On analysis of QoL among different JIA categories [Figure 3], parents' and child self-report showed better scores in children of sJIA in almost all individual domains as well as in summary and global scores followed by pJIA.
|Figure 3: Comparison of scores between juvenile idiopathic arthritis subtypes|
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Children from lower middle-class backgrounds scored better compared to that from the lower class in almost all domains, it was statistically significant in parents' physical, school, psychosocial, and global reports [Figure 4]. Children of parents with at least secondary level of education fared much better in almost all domains compared of children of less-educated/illiterate parents [Figure 5].
On trying to correlate the QoL with age, there was an overall and individual good positive correlation with HRQoL with increasing age in all domains of parent score while there was no correlation with age in child score. Both child self-assessment and parent scores were better with increasing BMI in almost all domains as well as in overall scores. The QoL improved with the increasing duration of the disease and with appropriate therapy both from the child's and parent's perspective. There was a remarkable negative correlation between disease activity levels and QoL in all domains both in child and parent scores which means children with more active disease had worse QoL than those in whom the disease was well in control [Table 2]. Overall, there was no significant difference in child self-assessment and parent proxy report scores in any of the domains [Table 3].
|Table 2: Correlation of various demographic, clinical and treatment related characteristics with health related quality of life scores|
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| Discussion|| |
Although the World Health Organization defined health taking into account the social and mental aspects also many decades ago, health outcomes of many diseases including chronic ones have traditionally been measured using measures of morbidity or mortality. However, as medical and public health advances have led to cures and or at least better controls of existing diseases with delayed mortality, it was logical that those who measure health outcomes would begin to assess the population's health not only on the basis of saving lives but also in terms of improving the quality of them. Hence, quite understandably, some attempts had been made in the latter part of the last millennium to assess the QoL in children suffering from chronic diseases, with some publications on this aspect of patient management.,,, Few studies have also been published on JIA but mostly from developed countries,,,,, like UK, Sweden, etc., the findings of which cannot always be extrapolated to a developing country setting because of socioeconomic, cultural, infrastructural, and resource differences.
Most of the studies hitherto published from India on chronic rheumatological conditions focus on the somatic aspect of the disease based on various scoring. Only a few have tried to include psychometric aspects as multidimensional evaluation of the disease activity. AS JIA is a chronic disease, it has every potential to adversely affect the social, emotional, and functional QoL in a child during his actively growing years not only because of the joint-related functional incapacitation but also the associated cardiovascular, physical, and psychological morbidities such as sleep disturbances, anxiety, fatigue, depression, etc. Holistic management of chronic diseases mandates that these aspects of the disease should also be taken care of rather than focusing only on relieving somatic symptoms of the disease.
In absence of a truly representative HRQoL reference population nomogram from the Indian sub-continent, we used PedsQL 4.0 as some studies published from India,, report better applicability of this tool in the setting of Indian children in the setting of both healthy children in school and also in certain chronic diseases like thalassemia.
On analysis of QoL in subtypes of JIA, children with oJIA scored lowest which could be due to the fact that although the oJIA children were relatively less in number, all of them had been referred from the peripheral hospital because of their severe and difficult to manage joint manifestations. The QoL improved with the passage of time in all domains both from both childrens' and parents' perspectives. It could be due to better understanding and adjustment to the disease process and better coping with a chronic illness. Improvement on QoL with the duration of therapy and negative correlation with disease activity score of DAS is quite understandable that as disease gets controlled with treatment, QoL improves.
[Table 4] compares the main inferences of this study with some other studies published previously, although the tools used in these studies are different and the results may not be truly comparable. Shaw et al. published a study on HRQol in older children with JIA (11–17 years) using a different tool (Juvenile Arthritis Quality of Life Questionnaire) significantly inferred that HRQol was significantly less optimal particularly in the physical domain. Furthermore, most of the older adolescents reported psychometric abnormalities with around 40% reporting frustration and 60% reporting depression. In a study by Seid et al. from Cinncinnati Children's Hospital, it was seen that in patients of polyarticular JIA even when the disease was well controlled with no or minimal clinical symptoms, the HRQol was suboptimal. It stresses upon the need for monitoring QoL.
|Table 4: Comparison of main inferences with that of some previously published studies|
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In our study, there was no significant discrepancy between child self-report and parent scores, although previous studies have pointed out certain discrepancies. Qadeer et al. on children with chronic disorders including asthma, diabetes, epilepsy and JIA using KIDSCREEN-27 showed a significant discrepancy between child and parent reporting on QoL when newly diagnosed which tended to flatten out with duration of time when child and parent reporting seemed to be more in agreement. A review by Upton et al. on parent-child agreement across pediatric HRQoL instruments concludes that the clinical significance of the actual difference between child and parent reporting of HRQOL is unclear. It also adds that parents may vary in their awareness and tolerance of children's health.
This study could portray some of the important aspects of QoL of children with JIA from a developing country setting, although it has its' share of limitations. Firstly, the study was conducted in a single center tertiary care hospital where the patient profile may not be a true representation of the actual disease profile in the community due to various factors such as referral bias, etc. As ours is a government institute where treatment is provided free of cost, the socioeconomic profile of the patients' families tends to be towards the poorer side as patients having better means prefer medical institutes in the private sector. Furthermore, separate information on the rural or urban background was not gathered.
Second, it was a single contact questionnaire-based study. Follow-up study over duration of time would have been more informative about the change in QoL as a result of clinical intervention. Furthermore, a follow-up study would have given us a more reliable information on drug compliance and defaulting. Third, we have used only a generic instrument but no specific instrument for measuring the QoL in children with JIA. And finally, in the absence of a control arm, we do not have any data regarding baseline HRQoL scores of healthy children constituting the same demographic profile from eastern India.
| Conclusion|| |
This study though small and hospital based, does give certain important insights into possible factors on which QoL of children suffering from JIA depend. Male gender, older age, systemic JIA, lower-middle socio-economic background compared to lower class, literacy at least up to the secondary level in parents, better BMI, better disease activity scores, and longer duration of disease and therapy correlated positively with better QoL scores in almost all domains. Child self-assessment score and parents' proxy report scores were comparable across all domains and also overall scores.
This study can be taken as a preliminary reference study from Eastern India based upon which larger community-based multicentric studies with healthy controls could be undertaken to arrive at more robust conclusions and help health agencies in formulating strategies to improve the HRQoL in such children.
Financial support and sponsorship
Conflicts of interest
There are no conflicts of interest.
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[Figure 1], [Figure 2], [Figure 3], [Figure 4], [Figure 5]
[Table 1], [Table 2], [Table 3], [Table 4]