Wendy Thomson

Disease activity and disability in children with juvenile idiopathic arthritis one year following presentation to paediatric rheumatology. Results from the Childhood Arthritis Prospective Study.

Objective. Inflammatory arthritis in childhood is variable in terms of both presentation and outcome. This analysis describes disease activity in children with juvenile idiopathic arthritis (JIA) during the first year following presentation to a paediatric rheumatologist and identifies predictors of moderate to severe disability [defined using a Childhood HAQ (CHAQ) score ⩾0.75] at 1 year. Methods. The Childhood Arthritis Prospective Study recruits children <16 years with new inflammatory arthritis persisting for ⩾2 weeks from five UK tertiary referral centres. Demographics, disease features, joint count, CHAQ, physicians global assessment, parents general evaluation of well-being (PGE), ESR and treatment, are collected at first presentation, 6 months and then yearly. Independent predictors of CHAQ ⩾0.75 at 1 year in children diagnosed with JIA were identified using multivariable logistic regression models. Results. Seven hundred and forty children with JIA were included; median age at presentation 7.6 years, 64% girls. During the first year, 85% received NSAIDs, 70% IA corticosteroids, 47% MTX and 27% systemic steroids (oral or i.v.). Median presenting CHAQ score was 0.63 and decreased to 0.25 at 1 year; 32% had CHAQ ⩾0.75 at 1 year. The strongest predictor of CHAQ ⩾0.75 at 1 year was CHAQ ⩾0.75 at presentation (odds ratio 3.92; 95% CI 2.17, 7.09). Additional predictors included female gender and higher PGE Conclusion. Although CHAQ score improved in most children, the strongest predictor of persistent disability at 1 year was moderate to severe disability at first presentation. Follow-up beyond 1 year will assess whether CHAQ at presentation will continue to be a predictor of future poor outcome.

Association between duration of symptoms and severity of disease at first presentation to paediatric rheumatology: results from the Childhood Arthritis Prospective Study

Objectives. To study the association between disease severity at first presentation to paediatric rheumatology (PRh) and length of time since symptom onset in children recruited to the Childhood Arthritis Prospective Study. Methods. Children ≤16 yrs with inflammatory arthritis persisting ≥2 weeks were recruited from five UK hospitals. Data including demographics, disease features, Childhood Health Assessment Questionnaire (CHAQ), physician and parent global assessment and blood tests were collected at the first appointment with PRh (baseline). The association between symptom duration (defined as time from first reported symptom onset to presentation at PRh) and baseline disease characteristics was evaluated using non-parametric descriptive statistics and multivariable logistic regression analyses. Results. Five hundred and seven children (65% female) were included: median age at onset was 6.8 yrs. Two hundred and thirty-three had oligoarthritis, 68 had RF-negative polyarthritis, 27 had systemic onset arthritis and 29 had arthritis that was not JIA. The median symptom duration was 4.6 months. Median symptom duration was shortest for children presenting with systemic arthritis (1.6 months) and longest for those with PsA (8.6 months). Children with a longer duration of symptoms were older and had higher median active joint counts but lower median ESR. Symptom duration did not correlate with CHAQ score at presentation. Conclusions. Children who have systemic arthritis had the shortest delay to PRh presumably because they are profoundly unwell. Children with joint pain/stiffness but normal ESR had longer delays suggesting that if blood tests do not indicate inflammation, the diagnosis of JIA may be overlooked.

Costing juvenile idiopathic arthritis: examining patient-based costs during the first year after diagnosis.

Objectives. There are few data on the treatment patterns and associated cost of treating children with inflammatory arthritis including juvenile idiopathic arthritis (JIA), in the short or long term. The aim of this study was to obtain patient-based costs for treating children with JIA in the UK, in the first year from diagnosis and from the secondary health care payer perspective. Methods. The Childhood Arthritis Prospective Study (CAPS) is an ongoing longitudinal study recruiting children with inflammatory arthritis from four UK hospital centres. Included children are newly diagnosed, ≤16 years old with inflammatory arthritis of one or more joints, which has persisted for at least 2 weeks. Health service resource use data were collected as part of routine clinical care at study entry, 6 months and 1 year. Reference unit costs were applied to these data and the cost of treatment per child calculated for the first year from diagnosis. Results. A total of 297 children attended a 12-month follow-up visit. The mean annual total cost per child was £1649 (s.d. £1093, range £401–£6967). The highest cost component was for appointments with paediatric rheumatologists. Mean total costs were highest for children with enthesitis-related, systemic JIA or extended oligoarthritis. Conclusions. In the first 12 months after diagnosis, children with all JIA disease subtypes consume large, but highly variable quantities of health service resources. Individual patient costs are required to reflect the wide variation in cost between patients and allow appropriate recouping of costs for contracted services and for assessing the economic impact of interventions.

Trends in paediatric rheumatology referral times and disease activity indices over a ten-year period among children and young people with Juvenile Idiopathic Arthritis: results from the childhood arthritis prospective Study

Objectives. The medical management of JIA has advanced significantly over the past 10 years. It is not known whether these changes have impacted on outcomes. The aim of this analysis was to identify and describe trends in referral times, treatment times and 1-year outcomes over a 10-year period among children with JIA enrolled in the Childhood Arthritis Prospective Study. Methods. The Childhood Arthritis Prospective Study is a prospective inception cohort of children with new-onset inflammatory arthritis. Analysis included all children recruited in 2001–11 with at least 1 year of follow-up, divided into four groups by year of diagnosis. Median referral time, baseline disease pattern (oligoarticular, polyarticular or systemic onset) and time to first definitive treatment were compared between groups. Where possible, clinical juvenile arthritis disease activity score (cJADAS) cut-offs were applied at 1 year. Results. One thousand and sixty-six children were included in the analysis. The median time from symptom onset and referral to first paediatric rheumatology appointment (22.7–24.7 and 3.4–4.7 weeks, respectively) did not vary significantly (∼20% seen within 10 weeks of onset and ∼50% within 4 weeks of referral). For oligoarticular and polyarticular disease, 33.8–47 and 25.4–34.9%, respectively, achieved inactive disease by 1 year, with ∼30% in high disease activity at 1 year. A positive trend towards earlier definitive treatment reached significance in oligoarticular and polyarticular pattern disease. Conclusion. Children with new-onset JIA have a persistent delay in access to paediatric rheumatology care, with one-third in high disease activity at 1 year and no significant improvement over the past 10 years. Contributing factors may include service pressures and poor awareness. Further research is necessary to gain a better understanding and improve important clinical outcomes.

Juvenile-onset inflammatory arthritis: a study of adolescents’ beliefs about underlying cause

Objective. Patients’ beliefs regarding the cause of illness may influence treatment adherence and long-term outcome. Little is known of adolescents’ beliefs regarding the cause of JIA. This study aims to identify adolescents’ beliefs about the underlying cause of their arthritis at first presentation to the paediatric rheumatology department. Methods. One hundred and twenty-two adolescents aged ≥11 years participating in the larger prospective Childhood Arthritis Prospective Study, an inception cohort of childhood-onset inflammatory arthritis, were asked to complete a questionnaire regarding underlying beliefs about their arthritis. The top-listed causes were identified, and associations between beliefs and characteristics of the adolescents and their arthritis were compared across the different causal beliefs. Results. The most common causal beliefs were genetics (27.1%), the immune system (21.3%), accident or injury (15.6%) and infection (13.1%). Association between causal beliefs and gender, disease duration, International League Against Rheumatism subtype and source of referral was observed, although small numbers prevented robust statistical comparisons. Conclusion. This first report on adolescents’ beliefs about the cause of their juvenile arthritis found the most common causal beliefs to be related to genes or the immune system. Brief assessments of adolescents’ beliefs at presentation will enable providers to modify or adapt potentially unhelpful beliefs and provide age-appropriate information regarding arthritis.

Minimal Disease Activity in a Clinical Cohort of Children with Juvenile Idiopathic Arthritis: Results from the Childhood Arthritis Prospective Study

Background: Children and young adults with JIA have increased levels of poor oral hygiene and dental decay [1]. Periodontitis and types of arthritis are linked by similar components of blood cytokine profiles. Good dental health can be directly affected in JIA patients due to physical limitations in upper limb movements making brushing and flossing teeth difficult. An important factor in oral care is good dental hygiene and access to dental health practitioners. NHS advice is that all children should be reviewed by a dentist annually and be offered both sealant of their teeth and fluoride varnish at the appropriate time. Our aim was to establish if our patients had any barriers to accessing dental care. Methods: All patients (age 18 and under) diagnosed with JIA in the paediatric rheumatology clinic over a period of 3 months were asked to complete a dental care questionnaire. Parents completed the questionnaire for their children if necessary. Data were analysed using Excel. Results: 30 questionnaires were completed. Demographics were M:F 1:1.3, all children were diagnosed with JIA, average age 10.5 years with range 2–18. 27 children were registered with an NHS dentist with the exception of one child with a private dentist. 26 children had seen a dentist at least annually and one child in the past 2 years. 2 children, one aged 16, were not registered with a dentist because their parents didn’t think it was important. 11 children had 25 fillings in total, 9 of these children were not supervised during dental hygiene. 13 children admitted to drinking sugary drinks daily and had 16 dental fillings. None of the children admitted to smoking. Conclusions: Whilst our audit showed that most children were registered with a dentist and were reviewed annually, only 1 child had been offered sealant and fluoride varnish. The NHS advises that children with chronic medical conditions can be seen either by their NHS dentist or by the local Community specialist dental service which can be accessed by referral from their rheumatology department or NHS dentist. None of the children were seen by the specialist dental service. We have developed an information leaflet informing parents and children with JIA of the importance of dental health explaining the benefits of both sealant and fluoride for teeth.