Abstract
Background and Objectives
The functional disability status of Indian children with juvenile idiopathic arthritis is unidentified. In this cross-sectional study functional capacity of 60 juvenile idiopathic arthritis patients was assessed by the Childhood Health Assessment Questionnaire.
Methods
A total of 60 juvenile idiopathic arthritis patients aged ranges from 1 to 12 years were recruited from a teaching hospital in eastern India. A childhood health assessment questionnaire was used to assess the functional health of children. Pain, patient’s/parent’s global assessment of general well-being, and physician’s global assessment were assessed.
Results
Childhood health assessment questionnaire disability index for oligoarticular juvenile idiopathic arthritis differed significantly from polyarticular juvenile idiopathic arthritis (P < 0.001), systemic-onset juvenile idiopathic arthritis (P = 0.018) and undifferentiated juvenile idiopathic arthritis (P < 0.001). There was a good to a strong positive correlation between the childhood health assessment questionnaire disability index with pain score, patient’s/parent’s global assessment score, and physician global assessment score for the total juvenile idiopathic arthritis cohort. regarding juvenile idiopathic arthritis subtypes, significant correlations were noted between the childhood health assessment questionnaire disability index with the patient’s/parent’s global assessment and physician’s global assessment (except for enthesitis-related arthritis).
Conclusions
Assessment and documentation of the functional health status of juvenile idiopathic arthritis patients will improve the management of the disease.
Introduction
Juvenile idiopathic arthritis (JIA) is universal childhood morbidity. It is categorized into seven groups; each subtype is characterized by specific features, disabilities, and complications.[1] Studies revealed that up to 70% of JIA patients continue to claim disability and restriction of their activities.[2] High functional disability leads to poor academic performance and school absenteeism.[3] The Childhood Health Assessment Questionnaire (CHAQ) is perhaps the most practiced questionnaire in clinical studies to measure functional outcomes in JIA.[2,3] Study from India addressing functional disability in JIA patients is lacking. We addressed this lacuna.
Methods
We conducted this cross-sectional study in a teaching hospital in eastern India from February 2022 to January 2023, over a year. JIA (International League of Associations for Rheumatology (ILAR) 2001 criteria) patients, 1 to 12 years of age, and their primary caregivers were included. The presence of cognitive disability and significant comorbidity were excluded. Functional health status was assessed by CHAQ which gives a disability index (DI). Pain, Patient’s/Parent’s Global Assessment (PtGA) of general well-being, and Physicians Global Assessment (PGA) were assessed. The study protocol was endorsed by the Institutional Ethics Committee for clinical research (MC/KoL/IEC/Non-Span/368/11–2016). Data were collected by a single investigator using a pretested structured interview schedule. To make the questionnaire user-friendly, CHAQ was translated (from English) into Bengali and Hindi languages with an adaption to Indian culture and lifestyle. Discrepancies in translation were resolved through consensus and the questionnaire was validated through pilot testing. Data have been summarized using descriptive statistics. Multiple JIA subgroups were compared by using the Kruskal-Wallis analysis of variance. Dunn’s test was used to pinpoint which pairwise differences were statistically significant. The strength of the association between numerical variables was quantified by Spearman’s rank correlation coefficient. In all comparisons, statistical significance was inferred if the p-value was < 0.05.
Results
Out of 70 (10 excluded; non-availability of consent = 3, lack of defined caregiver = 4, presence of disabling comorbidity = 30), 60 children (53.5% males) and their caregivers (93.3% females, 86.7% mothers) were interviewed. The mean age of patients and caregivers was 8.5 and 35.9 years, respectively. JIA subtypes encountered were oligoarticular (28.3%), polyarticular (23.3%), systemic onset JIA (SoJIA; 23.3%) undifferentiated (16.7%), and enthesitis-related arthritis (ERA; 8.3%). Disease burden parameters of patients in different JIA subgroups are summarized in Table 1. CHAQ DI for oligoarticular JIA differed significantly from polyarticular JIA (P < 0.001), SoJIA (P = 0.018), and undifferentiated JIA (P < 0.001). There was a good to strong positive correlation of CHAQ DI with pain score, PtGA score, and PGA score for the total JIA cohort. Regarding JIA subtypes, significant correlations were noted between CHAQ DI and PtGA (except ERA) and PGA (except ERA and undifferentiated JIA). However, for individual subtypes, correlations between disability index and pain scores were weak.
Summary of disease burden parameters for Juvenile Idiopathic Arthritis cases.
| Parameter | Oligoarticular JIA (n = 17) | Polyarticular JIA (n = 14) | Systemic onset JIA (n = 14) | Undifferentiated JIA (n = 10) | P-value |
|---|---|---|---|---|---|
| CHAQ-DI | < 0.001 | ||||
| Range | 0.80–2.00 | 1.80–2.50 | 1.70–2.20 | 0.60–2.70 | |
| Mean ± SD | 1.08 ± 0.28 | 2.24 ± 0.22 | 1.94 ± 0.18 | 2.11 ± 0.65 | |
| Median (IQR) | 1.00 (0.90–1.10) | 2.20 (2.20–2.40) | 1.95 (1.80–2.10) | 2.30 (2.00–2.60) | |
| Pain Score | < 0.001 | ||||
| Range | 1.50–6.00 | 5.00–7.50 | 5.00–7.10 | 3.00–8.00 | |
| Mean ± SD | 3.58 ± 1.20 | 6.56 ± 0.69 | 6.25 ± 0.62 | 5.88 ± 1.34 | |
| Median (IQR) | 3.60 (2.60–4.60) | 6.65 (6.00–7.00) | 6.35 (6.00–6.70) | 5.95 (5.00–6.90) | |
| PtGA Score | < 0.001 | ||||
| Range | 0.50–03.00 | 5.00–8.50 | 6.70–8.50 | 4.00–7.20 | |
| Mean ± SD | 1.82 ± 0.73 | 6.99 ± 0.87 | 7.60 ± 0.52 | 6.20 ± 0.88 | |
| Median (IQR) | 1.90 (1.05–2.55) | 6.95 (6.70–7.70) | 7.65 (7.10–8.00) | 6.40 (5.90–6.80) | |
| PGA Score | < 0.001 | ||||
| Range | 0.50–02.50 | 5.00–8.50 | 6.50–8.00 | 3.00–7.00 | |
| Mean ± SD | 1.73 ± 0.53 | 6.80 ± 1.01 | 7.37 ± 0.45 | 5.70 ± 1.20 | |
| Median (IQR) | 1.50 (1.50–2.00) | 7.00 (5.70–7.50) | 7.30 (7.00–7.70) | 6.00 (5.50–6.70) |
IQR, Interquartile range; SD, Standard deviation; JIA, Juvenile idiopathic arthritis; CHAQ-DI, Childhood Health Assessment Questionnaire Disability Index; PtG , patient’s/ parent’s global assessment; PGA, physician global assessment. Pain Score, PtGA Score and PGA Score were all recorded on 10 cm visual analog scales. P value in the last column is from multiple comparison with Kruskal-Wallis one-way analysis of variance.
Discussion
Polyarticular JIA imposed the highest functional disability in terms of CHAQ DI when various JIA subgroups were compared, whereas patients with oligoarticular JIA had the lowest disability. This observation is consistent with that of previous studies.[3–6] A French study documented that ERA patients had the worst scores for both pain and well-being on the visual analogue scale (VAS) scale. Because of differences in JIA disease activity, subtype distribution, and assessment methodology, precise comparison with pre-existing studies is not always possible.[4] In JIA subtypes, analysis significant correlations were noted between CHAQ DI and PtGA (except ERA) and PGA (except ERA and undifferentiated JIA). However, for individual subtypes, correlations between
DI and pain score were weak. This pattern broadly matches studies from other countries such as France,[4] Canada,[7] and Thailand.[3] There are minor conflicts with previous studies regarding CHAQ DI correlation with other functional health parameters and physician-assessed disease activity (i.e., PGA) as the degree of correlation varies across studies.[7] Contrary to this study, Boiu et al[4] reported a good correlation between CHAQ DI and VAS pain score within each JIA category.
However, this study has its share of limitations. It was a single tertiary care center-based study with a limited sample size and children up to twelve years of age were included.
Conclusion
In our study significant correlations were noted between CHAQ DI and PtGA (except ERA) and PGA (except ERA and undifferentiated JIA).
The work is a pioneering effort in documenting the functional health status of JIA patients in Indian scenarios. Assessment and documentation of the functional health status of juvenile idiopathic arthritis patients will improve the management of the disease.
Funding statement: The authors received no financial support for the research.
Acknowledgement
The authors would like to thank the participants and their families who gave their consent and collaborated in this study. In addition, we are also thankful to Multidisciplinary Research Unit, Medical College Kolkata for their laboratory support.
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Author contributions
The conception of the study, reviewing the draft, study design, literature search: R. Mondal; Data collection, making draft: D. Datta; Literature search, review draft, interpretation & analysis: M. Khatun; Draft, statistics, literature search: B. Bankura; Study design, review draft, and interpretation: M. Sarkar; Statistics, making the draft, literature search: A. Hazra; Reviewing the draft, study interpretation, literature search: D. Ivan M; Reviewing the draft, study interpretation: Manab Nandy.
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Informed Consent
Written consent from parents/caregivers/legal guardians was taken before the conduction of the study.
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Ethical approval
Ethical clearance obtained from the Institutional Ethics Committee with Ref No. MC/KoL/IEC/Non-Span/368/11-2016, Dated-19/11/16.
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Conflict of interest
The authors declared no conflicts of interest.
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Data availability statement
No additional data related to this research is available.
References
[1] Petty RE, Southwood TR, Manners P, et al. International League of Associations for Rheumatology classification of juvenile idiopathic arthritis: second revision, Edmonton, 2001. J Rheumatol. 2004;31:390-392.Suche in Google Scholar
[2] Hyrich KL, Lal SD, Foster HE, et al. Disease activity and disability in children with juvenile idiopathic arthritis one year following presentation to paediatric rheumatology. Results from the Childhood Arthritis Prospective Study. Rheumatology (Oxford). 2010;49:116–122.10.1093/rheumatology/kep352Suche in Google Scholar PubMed PubMed Central
[3] Sontichai W, Vilaiyuk S. The correlation between the Childhood Health Assessment Questionnaire and disease activity in juvenile idiopathic arthritis. Musculoskeletal Care. 2018;16:339-344.10.1002/msc.1239Suche in Google Scholar PubMed
[4] Boiu S, Marniga E, Bader-Meunier B, et al. Functional status in severe juvenile idiopathic arthritis in the biologic treatment era: an assessment in a French paediatric rheumatology referral centre. Rheumatology (Oxford). 2012;51:1285-1292.10.1093/rheumatology/kes004Suche in Google Scholar PubMed
[5] Ruperto N, Levinson JE, Ravelli A, et al. Long-term health outcomes and quality of life in American and Italian inception cohorts of patients with juvenile rheumatoid arthritis. I. Outcome status. J Rheumatol. 1997;24:945-951.Suche in Google Scholar
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© 2024 Debadyuti Datta, Moksuda Khatun, Biswabandhu Bankura, Mihir Sarkar, Avijit Hazra, D Ivan M, Manab Nandy, Rakesh Mondal, published by De Gruyter on behalf of NCRC-DID.
This work is licensed under the Creative Commons Attribution 4.0 International License.
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