A recurring rollercoaster ride: A qualitative study of the emotional experiences of parents of children with juvenile idiopathic arthritis

Background: Despite the wealth of clinical research carried out in children with juvenile idiopathic arthritis (JIA), little is known about the emotional experiences of their parents. This article describes the predominant emotional experiences reported by parents of children with JIA in two Canadian cities. Methods: Research participants included 15 experienced parents and 8 novice parents (\u3c6 months since children\u27s JIA diagnosis). Their children were 2 to 16 years old with various JIA categories. A qualitative dataset including audio recordings and verbatim transcripts of three focus groups, and written reports of 59 reciprocal interviews (parents interviewing each other) were examined by a multidisciplinary research team following a four-step qualitative analytical process. Results: Parents of children with JIA experienced recurrent mixed negative and positive emotions that varied over time. Between disease onset and diagnosis, mounting anxiety, fear and confusion were the predominant emotions. Shortly after diagnosis there were shock, disbelief, and fear, with a sense of having being blindsided by the disease. At times of disease quiescence there was hope and gratitude, but also fatigue and frustration with ongoing treatment and fear of flares. During periods of increasing or ongoing symptoms there was admiration and sympathy for the courageous way children coped with JIA, as well as sorrow and frustration for ongoing pain and limitations. There were at times, frustration and indignation with peers and teachers unable to understand the child\u27s fluctuations in physical activity and schoolwork. Throughout the disease, parents felt an underlying anxiety and powerlessness. Conclusions: Parents of children with JIA described complex emotional journeys akin to the recurring ups and downs of rollercoaster rides, instead of ordered emotional phases ending in resolution. This has implications for healthcare providers who need to be aware of the complexity of these emotional journeys to support parents more effectively, thereby helping improve patient outcomes

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ABSTRACT. Objective. The Research on Arthritis in Canadian Children Emphasizing Outcomes (ReACCh Out) cohort is a prospective inception cohort of patients with newly diagnosed juvenile idiopathic arthritis (JIA) seen in 16 Canadian pediatric rheumatology (PR) centers. We used data from this cohort to explore factors associated with longer time from symptom onset to the first visit to PR, and with longer time from first visit to a diagnosis of JIA. Methods. We included children enrolled in ReACCh Out within 6 months of JIA diagnosis, for whom the dates of symptom onset and first PR visit were recorded. We used Cox proportional hazard modeling to investigate the effects of history, physical examination, and laboratory evaluation on the interval from JIA symptom onset to first PR assessment. Results. In total, 319 children from the cohort were included. Having a fever (hazard ratio 1

The Effect of Guselkumab on Work Productivity in Biologic-Naïve Patients with Active Psoriatic Arthritis Through Week 52 of the Phase 3, Randomized, Placebo-Controlled DISCOVER-2 Trial

Introduction: The phase 3 DISCOVER-2 trial evaluated the effect of guselkumab on impaired work productivity and nonwork activity in biologic-naïve patients with psoriatic arthritis (PsA). Methods: Adults with active PsA were randomized (1:1:1) to guselkumab 100 mg every 4 weeks (Q4W), guselkumab 100 mg at weeks 0 and 4 and then every 8 weeks (Q8W), or placebo (with crossover to guselkumab Q4W at week 24). Least squares mean change from baseline in Work Productivity and Activity Impairment Questionnaire for PsA (WPAI-PsA) domains and employment were assessed by treatment group. Multivariate analysis of data from weeks 0 through 24 assessed independent associations between PsA clinical features and WPAI-PsA domains. Results: In total, 738 patients were evaluated (guselkumab Q4W n = 245; guselkumab Q8W n = 248; placebo n = 245). At week 24, improvements (reduced impairment) in presenteeism (Q4W −20.1%, Q8W −19.6%, placebo −10.5%), work productivity (Q4W −20.1%, Q8W −19.2%, placebo −10.6%), and nonwork activity (Q4W −20.5%, Q8W −21.2%, placebo −9.9%) were greater in guselkumab-treated versus placebo-treated patients. At week 52, following placebo crossover at week 24, improvements were similar among groups. Baseline absenteeism was minimal and did not change in any group. By week 52, 23.1–25.9% of guselkumab-treated patients who were unemployed at baseline were employed. All WPAI-PsA domains were positively associated with C-reactive protein level, fatigue, and pain. All domains except absenteeism were positively associated with enthesitis and Psoriasis Area and Severity Index score. Age was negatively associated with presenteeism and work productivity loss, female sex and tender joint count were positively associated with nonwork activity impairment, and dactylitis was positively associated with presenteeism. Conclusion: Both guselkumab regimens reduced work productivity loss and nonwork activity impairment in patients with active PsA. Association of work productivity loss and nonwork activity impairment with PsA joint and skin features suggests that improvement in both features is beneficial for optimizing improved work productivity loss and nonwork activity impairment. Trial registration: ClinicalTrials.gov identifier, NCT03158285

The prevalence of systemic autoimmune rheumatic diseases in Canadian pediatric populations: administrative database estimates

CI 17.9, 29.2). SARDs were more common in females than in males across all provinces. There was a slightly higher prevalence among those living in urban compared to rural areas of Alberta (rate difference 14.4, 95 % CI 8.6, 20.1) and Saskatchewan (rate difference 13.8, 95 % CI 1.0, 26.6). Our results provide population-based prevalence estimates of pediatric SARDs in four Canadian provinces. Keywords Pediatric rheumatic diseases · Systemic autoimmune rheumatic diseases · Epidemiology · Disease prevalence Abstract To estimate systemic autoimmune rheumatic disease (SARD) prevalence using administrative data for pediatric populations in four Canadian provinces. Physician billing claims and inpatient hospitalizations from Alberta, Manitoba, Quebec, and Saskatchewan were used to define cases aged ≤18 years with a SARD diagnosis code in: one or more hospitalization, two or more physician visits within 2 years and at least 2 months apart, or one or more physician visit to a rheumatologist. Estimates ranged from 15.9/100,000 in Quebec [95 % confidence interval (95 % CI) 14.1, 18.0] to 23.0/100,000 in Manitoba (95 % Rheumatology INTERNATIONA

Feasibility and safety of a 6-month exercise program to increase bone and muscle strength in children with juvenile idiopathic arthritis

Background: Arthritis in childhood can be associated with muscle weakness around affected joints, low bone mass and low bone strength. Exercise is recognized as an important part of management of children with juvenile idiopathic arthritis (JIA) but the exercise prescription to best promote bone and muscle health is unknown. We therefore aimed to: 1. assess feasibility and safety of a 6-month home- and group-based exercise program for children with JIA; 2. estimate the effect of program participation on bone mass and strength, muscle function and clinical outcomes and 3. determine if any positive changes in bone and muscle outcomes are maintained 6 months later. Methods: We recruited 24 children with JIA who were part of the Linking Exercise, Physical Activity and Pathophysiology in Childhood Arthritis (LEAP) study to participate in a 6-month home-based exercise program involving jumping and handgrip exercises, resistance training and one group exercise session per month. We assessed lumbar spine bone mass (dual energy X-ray absorptiometry), distal tibia and radius bone microarchitecture and strength (high-resolution peripheral quantitative computed tomography), muscle function (jumping mechanography, dynamometry) and clinical outcomes (joint assessment, function, health-related quality of life) at baseline, 6- and 12-months. Adherence was assessed using weekly activity logs. Results: Thirteen children completed the 6-month intervention. Participants reported 9 adverse events and post-exercise pain was rare (0.4%). Fatigue improved, but there were no other sustained improvements in muscle, bone or clinical outcomes. Adherence to the exercise program was low (47%) and decreased over time. Conclusion: Children with JIA safely participated in a home-based exercise program designed to enhance muscle and bone strength. Fatigue improved, which may in turn facilitate physical activity participation. Prescribed exercise posed adherence challenges and efforts are needed to address facilitators and barriers to participation in and adherence to exercise programs among children with JIA. Trial registration: Data of the children with JIA are from the LEAP study (Canadian Institutes of Health Research (CIHR; GRANT# 107535). http://www.leapjia.com/

Predicting which children with juvenile idiopathic arthritis will not attain early remission with conventional treatment: Results from the Reacch-out cohort

Objective. To estimate the probability of early remission with conventional treatment for each child with juvenile idiopathic arthritis (JIA). Children with a low chance of remission may be candidates for initial treatment with biologics or triple disease-modifying antirheumatic drugs (DMARD). Methods. We used data from 1074 subjects in the Research in Arthritis in Canadian Children emphasizing Outcomes (ReACCh-Out) cohort. The predicted outcome was clinically inactive disease for ≥ 6 months starting within 1 year of JIA diagnosis in patients who did not receive early biologic agents or triple DMARD. Models were developed in 200 random splits of 75% of the cohort and tested on the remaining 25% of subjects, calculating expected and observed frequencies of remission and c-index values. Results. Our best Cox logistic model combining 18 clinical variables a median of 2 days after diagnosis had a c-index of 0.69 (95% CI 0.67-0.71), better than using JIA category alone (0.59, 95% CI 0.56-0.63). Children in the lowest probability decile had a 20% chance of remission and 21% attained remission; children in the highest decile had a 69% chance of remission and 73% attained remission. Compared to 5% of subjects identified by JIA category alone, the model identified 14% of subjects as low chance of remission (probability \u3c 0.25), of whom 77% failed to attain remission. Conclusion. Although the model did not meet our a priori performance threshold (c-index \u3e 0.70), it identified 3 times more subjects with low chance of remission than did JIA category alone, and it may serve as a benchmark for assessing value added by future laboratory/imaging biomarkers

Glucocorticoid-related changes in body mass index among children and adolescents with rheumatic diseases

Objective To examine the temporal and dose-related effects of glucocorticoids (GCs) on body mass index (BMI) in children with rheumatic diseases. Methods Children initiating GCs for a rheumatic disease (n = 130) were assessed every 3 months for 18 months. BMI, weight, and height Z score trajectories were described according to GC starting dosage in prednisone equivalents: high (≥1.0 mg/kg/day), low (\u3c0.2 mg/kg/day to a maximum of 7.5 mg/day), and moderate (between high and low) dosage. The impact of GC dosing, underlying diagnosis, pubertal status, physical activity, and disease activity on BMI Z scores and on percent body fat was assessed with longitudinal mixed-effects growth curve models. Results The GC starting dose was high in 59% and moderate in 39% of patients. The peak BMI Z score was +1.29 at 4 months with high-dose GCs and +0.69 at 4.2 months with moderate-dose GCs (P \u3c 0.001). Overall, 50% (95% confidence interval 41-59%) of the children returned to within +0.25 SD of their baseline BMI Z score. Oral GC dose over the preceding 3 months was the most significant determinant of BMI Z score and percent body fat. The proportion of days in receipt of GCs, disease activity, and a diagnosis of systemic-onset juvenile idiopathic arthritis were also associated with BMI Z scores. The correlation between changes in BMI and changes in percent body fat was 0.09. Conclusion In children with rheumatic diseases starting moderate and high doses of GCs, BMI Z scores peaked at 4 months, and only half returned to within +0.25 SD of their baseline BMI Z score after 18 months. Copyright © 2013 by the American College of Rheumatology

The risk and nature of flares in juvenile idiopathic arthritis: Results from the ReACCh-Out cohort

Objective To describe probabilities and characteristics of disease flares in children with juvenile idiopathic arthritis ( JIA) and to identify clinical features associated with an increased risk of flare. Methods We studied children in the Research in Arthritis in Canadian Children emphasizing Outcomes (ReACCh-Out) prospective inception cohort. A flare was defined as a recurrence of disease manifestations after attaining inactive disease and was called significant if it required intensification of treatment. Probability of first flare was calculated with Kaplan-Meier methods, and associated features were identified using Cox regression. Results 1146 children were followed up a median of 24 months after attaining inactive disease. We observed 627 first flares (54.7% of patients) with median active joint count of 1, physician global assessment (PGA) of 12 mm and duration of 27 weeks. Within a year after attaining inactive disease, the probability of flare was 42.5% (95% CI 39% to 46%) for any flare and 26.6% (24% to 30%) for a significant flare. Within a year after stopping treatment, it was 31.7% (28% to 36%) and 25.0% (21% to 29%), respectively. A maximum PGA \u3e30 mm, maximum active joint count \u3e4, rheumatoid factor (RF)-positive polyarthritis, antinuclear antibodies (ANA) and receiving disease-modifying antirheumatic drugs (DMARDs) or biological agents before attaining inactive disease were associated with increased risk of flare. Systemic JIA was associated with the lowest risk of flare. Conclusions In this real-practice JIA cohort, flares were frequent, usually involved a few swollen joints for an average of 6 months and 60% led to treatment intensification. Children with a severe disease course had an increased risk of flare

Response to Treatment with Intravenous Golimumab or Infliximab in Rheumatoid Arthritis Patients: PROMIS Results from the Real-World Observational Phase 4 AWARE Study

Abstract Introduction To assess changes in the Patient-Reported Outcomes Measurement Information System (PROMIS®) outcomes related to social, mental, and physical well-being after approximately 1 year of intravenous (IV) golimumab or infliximab treatment in patients with rheumatoid arthritis (RA) using real-world evidence from AWARE. Methods AWARE was a prospective, noninterventional, multicenter, observational, U.S.-based phase 4 study of 1270 RA patients who initiated treatment with IV golimumab or infliximab. PROMIS-29 and PROMIS short form (SF) Fatigue 7a and Pain Interference 6b questionnaires were administered at baseline and infusions 2, 5, and 8 (approximately weeks  4, 28, and 52 for IV golimumab and weeks  2, 22, and 46 for infliximab). Mean changes from baseline in all PROMIS-29 domains and respective SFs and response rates for achieving ≥ 3, ≥ 5, or ≥ 10-point improvements were determined. Results Among all patients, baseline mean ± SD PROMIS T-scores were consistent between treatment groups and indicated worse physical function (38.2 ± 6.8 IV golimumab, 38.0 ± 6.9 infliximab), more pain interference (63.0 ± 7.6 IV golimumab, 63.9 ± 7.8 infliximab), and greater fatigue (58.4 ± 9.9 IV golimumab, 59.4 ± 10.0 infliximab) in these patients vs the general U.S. population (T-score = 50). Through the 8th infusion of either treatment, IV golimumab- and infliximab-treated patients achieved meaningful improvements (≥ 3-point improvement in T-scores) in all PROMIS-29 domains and respective SFs, and the proportions of patients with ≥ 3, ≥ 5, or ≥ 10-point improvements in T-scores increased from infusion 2 through infusion 8. Conclusions RA patients treated with IV golimumab or infliximab achieved comparable improvements across social, mental, and physical well-being PROMIS measures. Additionally, PROMIS detected meaningful clinical changes in patient-reported outcomes in both treatment groups. ClinicalTrials.gov registration number NCT02728934