Predicting Which Children with Juvenile Idiopathic Arthritis Will Have a Severe Disease Course: Results from the ReACCh-Out Cohort

Guzman, Jaime; Henrey, Andrew; Loughin, Thomas M.; Berard, Roberta; Shiff, Natalie J.; Juřenčák, Roman; Benseler, Susanne M.; Tucker, Lori B.

doi:10.3899/jrheum.160197

Cited by 47 publications

(67 citation statements)

References 22 publications

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“…Our results on early pain intensity with a mean VAS pain score of 3.0 are consistent with a recent cross‐sectional study in children and adolescents with JIA from the southeastern region of the US, showing a mean VAS pain score of 2.6 . Our results on pain intensity at the 8‐year follow‐up appear to be lower compared to other studies . Those studies are, however, skewed to the severe end of the JIA spectrum, whereas our population‐based study included the full disease spectrum.…”

Section: Discussionsupporting

confidence: 89%

Early Self‐Reported Pain in Juvenile Idiopathic Arthritis as Related to Long‐Term Outcomes: Results From the Nordic Juvenile Idiopathic Arthritis Cohort Study

Arnstad

Rypdal

Peltoniemi

et al. 2019

Arthritis Care & Research

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Section: Discussionsupporting

confidence: 89%

Early Self‐Reported Pain in Juvenile Idiopathic Arthritis as Related to Long‐Term Outcomes: Results From the Nordic Juvenile Idiopathic Arthritis Cohort Study

Arnstad

Rypdal

Peltoniemi

et al. 2019

Arthritis Care & Research

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“…Likewise, this study investigated adolescent‐onset disease only. Since the greatest change in functional ability is observed shortly after diagnosis of JIA , it could be expected that adolescents with childhood‐onset JIA experience relatively mild functional disability similar to their adolescent‐onset peers, thus lending the results of this study generalizable to both groups of young individuals in adolescence. In addition, there may have been value in also comparing self‐completed C‐HAQ forms using the proxy‐completed version of the C‐HAQ.…”

Section: Discussionmentioning

confidence: 85%

“…Likewise, this study investigated adolescent-onset disease only. Since the greatest change in functional ability is observed shortly after diagnosis of JIA (20), it could be expected that adoles- Table 4. Receiver operating characteristics comparing the proxy-completed C-HAQ with the adolescent-specific C-HAQ and the HAQ over the first year following initial presentation* * AUC = area under the curve; 95% CI = 95% confidence interval; C-HAQ = Childhood Health Assessment Questionnaire.…”

Section: Discussionmentioning

confidence: 99%

Comparing Proxy, Adolescent, and Adult Assessments of Functional Ability in Adolescents With Juvenile Idiopathic Arthritis

Shoop-Worrall

Hyrich

Verstappen

et al. 2020

Arthritis Care & Research

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Objective In pediatric research, investigators rely on proxy reports of outcome, such as the proxy‐completed Childhood Health Assessment Questionnaire (C‐HAQ), to assess function in juvenile idiopathic arthritis (JIA). As children mature, they may self‐complete the adult HAQ or the unvalidated adolescent‐specific C‐HAQ. It is unclear how these measures compare and whether they are directly interchangeable. The present study was undertaken to compare agreement between the proxy‐completed C‐HAQ, adolescent‐specific C‐HAQ, and the HAQ at initial presentation to pediatric rheumatologic care and 1 year following the first presentation in adolescents with JIA. Methods Adolescents ages 11–17 years participating in the Childhood Arthritis Prospective Study (CAPS), a UK multicenter inception cohort, were included. In a CAPS substudy, adolescents self‐completed the adolescent‐specific C‐HAQ and the HAQ, and proxies simultaneously completed the proxy‐completed C‐HAQ at baseline and 1 year. Correlation and agreement between scores were assessed at baseline. Agreement and ability to similarly classify clinically important changes over time were assessed at 1 year following initial presentation to rheumatologic care. Results A total of 107 adolescents (adolescent‐specific C‐HAQ and HAQ) or their proxies (proxy‐completed C‐HAQ) had completed all 3 measures at baseline. Median age at diagnosis was 13 years, and 61% were female. Although the 3 scores demonstrated strong correlations (r > 0.8), they were not completely interchangeable, with agreement ranging between 70% and 80%. There was similar agreement between the changes in scores between baseline and 1 year. Using proxy‐completed C‐HAQ minimum clinically important cutoffs, the adolescent‐specific C‐HAQ and the HAQ similarly classified 80% to 90% of adolescents as having improved or worsened. Conclusion While there is relatively high agreement and similar classification of change between HAQ and the 2 C‐HAQ scores, these are not completely interchangeable. This impacts the comparison of function when measured in different ways over the lifespan.

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“…The primary definition of remission was clinical inactive disease for at least 12 months while off treatment [14]. We also examined the model's ability to predict a severe disease course as defined by Guzman et al [7], based on cluster analysis of changes in pain, health related quality of life, number of active joints, medication requirements, and medication side effects over 5 years.…”

Section: Discussionmentioning

confidence: 99%

“…Using data from the Research in Arthritis in Canadian Children Emphasizing Outcomes (ReACCh-Out) Cohort, Guzman et al developed a clinical prediction model to predict a severe disease course that had a Cindex of 0.85 in internal validation in that cohort [7]. Using data from the Nordic Study Group of Pediatric Rheumatology (NoSPeR) cohort, Rypdal et al developed models to predict non-achievement of remission, functional disability, and articular damage 8 years after disease onset.…”

Section: Introductionmentioning

confidence: 99%

Validation of prediction models of severe disease course and non-achievement of remission in juvenile idiopathic arthritis part 2: results of the Nordic model in the Canadian cohort

Henrey

Rypdal

et al. 2020

Arthritis Res Ther

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Background: Validated clinical prediction models to identify children with poor prognosis at the time of juvenile idiopathic arthritis (JIA) diagnosis would be very helpful for tailoring treatments, and avoiding under-or overtreatment. Our objective was to externally validate Nordic clinical prediction models in Canadian patients with JIA. Methods: We used data from 513 subjects at the 3-year follow-up from the Research in Arthritis in Canadian Children emphasizing Outcomes (ReACCh-Out) cohort. The predicted outcomes were non-achievement of remission, severe disease course, and functional disability. The Nordic models were evaluated exactly as published and after fine-tuning the logistic regression coefficients using multiple data splits of the Canadian cohort. Missing data was handled with multiple imputation, and prediction ability was assessed with C-indices. C-index values > 0.7 were deemed to reflect helpful prediction. Results: Overall, 81% of evaluable patients did not achieve remission off medications, 15% experienced a severe disease course, and 38% reported disability (CHAQ score > 0). The Nordic model for predicting non-achievement of remission had a C-index of 0.68 (95% CI 0.62-0.74), and 0.74 (0.67-0.80) after fine-tuning. For prediction of severe disease course, it had a C-index of 0.69 (0.61-0.78), and 0.79 (0.68-0.91) after fine-tuning. The fine-tuned Nordic model identified 85% of the cohort as low risk for a severe disease course (< 20% chance) and 7% as high risk (> 60% chance). The Nordic model to predict functional disability had a C-index of 0.57 (0.50-0.63), and 0.51 (0.39-0.63) after fine-tuning. Conclusions: Fine-tuned Nordic models, combining active joint count, physician global assessment of disease activity, morning stiffness, and ankle involvement, predicted well non-achievement of remission and severe disease course in Canadian patients with JIA. The Nordic model for predicting disability could not predict functional disability in Canadian patients.

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Predicting Which Children with Juvenile Idiopathic Arthritis Will Have a Severe Disease Course: Results from the ReACCh-Out Cohort

Abstract: Children in this JIA cohort followed 1 of 4 disease courses and the probability of a severe disease course could be estimated with information available at diagnosis.

Cited by 47 publications

References 22 publications

Early Self‐Reported Pain in Juvenile Idiopathic Arthritis as Related to Long‐Term Outcomes: Results From the Nordic Juvenile Idiopathic Arthritis Cohort Study

Early Self‐Reported Pain in Juvenile Idiopathic Arthritis as Related to Long‐Term Outcomes: Results From the Nordic Juvenile Idiopathic Arthritis Cohort Study

Comparing Proxy, Adolescent, and Adult Assessments of Functional Ability in Adolescents With Juvenile Idiopathic Arthritis

Validation of prediction models of severe disease course and non-achievement of remission in juvenile idiopathic arthritis part 2: results of the Nordic model in the Canadian cohort

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