OP0278-PARE PROVIDING SUPPORT TO FAMILIES OF CHILDREN NEWLY-DIAGNOSED WITH CHILDHOOD ARTHRITIS: A PATIENT AND PARENT-LED PILOT STUDY TO DEVELOP AND ASSESS ‘A LITTLE BOX OF HOPE’ SUPPORT PACKS

Background:Juvenile Idiopathic Arthritis (JIA) is a heterogenous group of autoimmune disorders characterised by chronic joint inflammation, diagnosed in around 1 in 1,000 children and young people (CYP) under the age of 16. Delays in diagnosis are common [1], awareness is low, and paediatric rheumatological conditions have a considerable impact on young people and their families [2]. A lack of understanding amongst families of newly-diagnosed children leads to uncertainty and anxiety.Objectives:This patient and parent-led project developed a resource pack for parents of CYP newly-diagnosed with JIA, to provide information and support. Following a pilot, feedback from recipients was collated and analysed to help improve future provision.Methods:A young person with JIA identified the need for direct family support. Juvenile Arthritis Research (a UK charity) developed a unique pack of support information, containing resources for both children and their families - called A Little Box Of Hope. This included information about JIA and support services available for families, as well as Kipo (a children’s book about JIA) and accompanying finger puppet. Clinicians at one paediatric rheumatology centre provided information about the packs to newly diagnosed families, who then requested a free box to be posted to them.Following an initial pilot study, recipients were invited to complete a short online questionnaire and provide feedback to allow refinement of the provision.Results:Respondents were asked a series of questions, each on a scale of 1-5. Every respondent gave a score of 5 in response to “What do you think of the idea of A Little Box Of Hope?”Every parent of children under ten years old gave a score of 5 for every item when asked “How useful is each item in your Little Box Of Hope?”Respondents also gave free-text comments:* “It was very well thought out and I felt supported”* “I know so much more about JIA now than I did before. I cannot thank you enough.”* “It was extremely useful and made me feel supported during a very stressful time and this enabled me to support my son more effectively.”* “It made my daughter feel less alone.”Some parents of older children felt that some information specifically for teens would be useful, and a Teen pack is being developed.Conclusion:Recipients of A Little Box Of Hope have found the information useful and feel supported. Following the pilot study, we have developed My JIA, a booklet reviewed by a multi-disciplinary clinical team, with comprehensive information for families affected by JIA. A Teen pack, for children aged around 10 years or older, is being developed to provide targeted support to this group.The COVID-19 pandemic has adversely affected access to healthcare services, increasing the need for remote parent- and charity-provided support through A Little Box Of Hope.As such, we intend to expedite the roll-out of the project across the country building on the success of the pilot project.References:[1]McErlane F, Foster HE, Carrasco R, et al. 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. Rheumatology (Oxford) 2016;55(7):1225–34.[2]Foster HE, Scott C, Tiderius CJ, et al. Improving Musculoskeletal Health for Children and Young People - A “Call to Action”. Best Pract Res Clin Rheumatol 2020;34(5):101566.[3]Dejaco C, Alunno A, Bijlsma JWJ, et al. Influence of COVID-19 Pandemic on Decisions for the Management of People with Inflammatory Rheumatic and Musculoskeletal Diseases: A Survey among EULAR Countries. Ann Rheum Dis 2020;0:1-9.Disclosure of Interests:None declared

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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

Rheumatology ◽

10.1093/rheumatology/kew021 ◽

2016 ◽

Vol 55 (7) ◽

pp. 1225-1234 ◽

Cited By ~ 29

Author(s):

Flora McErlane ◽

Helen E. Foster ◽

Roberto Carrasco ◽

Eileen M. Baildam ◽

S. E. Alice Chieng ◽

...

Keyword(s):

Juvenile Idiopathic Arthritis ◽

Young People ◽

Prospective Study ◽

Disease Activity ◽

Paediatric Rheumatology ◽

Children And Young People ◽

Childhood Arthritis ◽

Activity Indices

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O002. Factors facilitating the self- and shared-management of Juvenile Idiopathic Arthritis

Rheumatology ◽

10.1093/rheumatology/keab723.001 ◽

2021 ◽

Vol 60 (Supplement_5) ◽

Author(s):

Simon Stones

Keyword(s):

Juvenile Idiopathic Arthritis ◽

Young People ◽

Paediatric Rheumatology ◽

The Self ◽

Self Management ◽

Management Support ◽

Children And Young People ◽

Realist Approach ◽

Shared Management ◽

Initial Question

Abstract Background Juvenile idiopathic arthritis (JIA) requires some form of lifelong management, with at least one third of children symptomatic in adulthood. Therefore, empowering children to competently self-manage their health and wellbeing across the lifecourse is logical, while supporting families in their shared-management role during childhood. However, there was a limited theoretical basis to the self- and shared-management of JIA across the lifecourse. The objective is to explore the factors facilitating the self- and shared-management of JIA using a realist approach to evaluation. Methods Guided by the Individual and Family Self-management Theory, a three-stage realist approach to evaluation was undertaken: 1) initial JIA self- and shared-management question theories were elicited from literature reviews and stakeholder insights [1]; 2) seven initial question theories were tested using teacher-learner cycle interviews with 20 participants; 3) findings were analysed using a theory-driven approach to thematic analysis, using deductive, inductive, and retroductive reasoning to extend or refute the initial question theories, in order to identify demi-regularities in the data. Results Six refined JIA self- and shared-management question theories emerged: 1) meaningful and bespoke self-management support across the life course for children and young people with JIA; 2) recognised and valued shared-management support for the families of children and young people with JIA, with autonomy in mind; 3) individual healthcare plans as a shared management communication tool to facilitate optimal management of JIA; 4) consistent recognition, value, and encourage of self- and shared-management support from the paediatric rheumatology multi-disciplinary team and associated professionals; 5) child, young-person, and family-focused paediatric rheumatology care and support services across the lifecourse; and 6) bespoke and inclusive approaches by education providers to enable children and young people with JIA to feel safe, supported, and able to fulfil their potential. Conclusion There is an increasing recognition of the importance of self- and shared-management of JIA and other paediatric-onset chronic conditions. However, there is a lack of an overall, cohesive approach to self- and shared-management between healthcare providers, education providers, and patient/parent organisations. The findings from this study illuminate the factors facilitating JIA self- and shared-management at individual, interpersonal, institutional and infrastructural levels, bearing relevance to individuals and organisations involved in caring for, and supporting children with JIA and their families.

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P20 Biosimilar use in children and young people with juvenile idiopathic arthritis in a real-world setting in the United Kingdom

Rheumatology ◽

10.1093/rheumatology/kez415.016 ◽

2019 ◽

Vol 58 (Supplement_4) ◽

Author(s):

Lianne Kearsley-Fleet ◽

Rebecca Adam ◽

Eileen Baildam ◽

Michael W. Beresford ◽

Helen E. Foster ◽

...

Keyword(s):

Juvenile Idiopathic Arthritis ◽

Young People ◽

Disease Activity ◽

Biologic Therapy ◽

Conflicts Of Interest ◽

Musculoskeletal Diseases ◽

Active Joint ◽

Children And Young People ◽

The United Kingdom

Abstract Background Most research concerning biosimilar use in musculoskeletal diseases has been conducted in adults. Little to no data regarding the use of biosimilars in children and young people with juvenile idiopathic arthritis (JIA) exist. Our aim was to describe the characteristics of children and young people with JIA starting biosimilars in the United Kingdom (UK) with musculoskeletal diseases. Methods The Biologics for Children with Rheumatic Diseases (BCRD) study is an ongoing prospective UK study recruiting children and young people with JIA starting biologic therapies other than etanercept originator (followed in a separate parallel study) from 2010 onwards. Baseline information is collected via questionnaires completed by the treating physician or affiliated clinical research nurse. Follow-up data including disease activity measures and changes in drug therapy are collected at six months, one year and annually thereafter. From May-2015, data has been captured on anti-TNF biosimilars in the UK. Results To 18 June 2019, 159 patients had started a biosimilar; 84 (53%) infliximab (2 products), 60 (38%) etanercept (2 products), and 15 (9%) adalimumab. Of these, 53 (33%) started the biosimilar as their first biologic therapy. Fifty (31%) patients switched from their originator, median (IQR) time on originator before switch was 2.2 (1.1-4.1) years, with most citing cost or hospital policy (n = 22) as the reason and these patients had low disease activity (median active joint count was zero) at point of switch. Fifty-six (35%) patients switched to a biosimilar from a non-originator biologic with 37 (66%) switching for efficacy reasons and 13 (23%) for safety reasons, with higher disease activity reported (median active joint count was three); most (n = 32) switching from adalimumab. Follow-up data was available in 143 patients; median follow-up time was 1.1 (0.7, 1.9) years. Sixty-seven patients switched to another biologic in this period. The majority (n = 38) switched to a non-originator biologic, predominantly adalimumab (n = 22) or tocilizumab (n = 14). Nineteen patients switched between infliximab biosimilars. Ten patients switched to the originator biologic (two due to inefficacy, four due to adverse events, four due to painful injections), although only four of these were on the originator prior to starting biosimilar; overall, in patients who switched from originator to biosimilar, 8% switched back to the originator. Conclusion Biosimilars in children and young people with JIA are being used as both first-line and subsequent-line biologic therapy. Patients starting a biosimilar from an originator product had lower disease activity and reported switching due to cost reasons, whilst patient starting a biosimilar from a non-originator were doing so due to inefficacy. Overall tolerance of biosimilar appears to be good so far, with few patients switching back to the originator. It is imperative that these patients continue to be followed within the registers so that effectiveness and safety of these treatments can be captured. Conflicts of Interest The authors declare no conflicts of interest.

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‘Snakes & Ladders’: factors influencing access to appropriate care for children and young people with suspected juvenile idiopathic arthritis – a qualitative study

Pediatric Rheumatology ◽

10.1186/s12969-021-00531-3 ◽

2021 ◽

Vol 19 (1) ◽

Author(s):

Tim Rapley ◽

Carl May ◽

Nicola Smith ◽

Helen E. Foster

Keyword(s):

Health Care ◽

Juvenile Idiopathic Arthritis ◽

Young People ◽

Health Professionals ◽

Health Care Professionals ◽

Paediatric Rheumatology ◽

Health Practitioner ◽

Children And Young People ◽

Appropriate Care ◽

Initial Symptoms

Abstract Background Many children and young people with juvenile idiopathic arthritis (JIA) experience delay in diagnosis and access to right care. The reasons for delay are multi-factorial and influenced by patient and family, clinician and organisational factors. Our aim was to explore the experiences of care, from initial symptoms to initial referral to paediatric rheumatology. Methods We analysed one-to-one and joint qualitative interviews with families of children with JIA (n = 36) presenting to a regional paediatric rheumatology service in the UK. We interviewed 51 family members (including mothers, fathers, patients, grandmothers and an aunt) and 10 health professionals (including orthopaedic surgeons, paediatricians, paediatric immunologist, General Practitioner and nurse) and a teacher involved in the care pathway of these JIA patients. Interviews were audio-recorded and analysed according to the standard procedures of rigorous qualitative analysis - coding, constant comparison, memoing and deviant case analysis. Results The median age of the children was 6 years old (range 1–17), with a spread of JIA subtypes. The median reported time to first PRh MDT visit from symptom onset was 22 weeks (range 4-364 weeks). Three key factors emerged in the pathways to appropriate care: (i) the persistence of symptoms (e.g. ‘change’ such as limp or avoidance of previously enjoyed activities); (ii) the persistence of parents help-seeking actions (e.g. repeat visits to primary and hospital care with concern that their child is not ‘normal’; iii) the experience and skills of health professionals resulting in different trajectories (e.g. no-real-concern-at-this-point or further-investigation-is-required). JIA was more likely to be considered amongst health practitioner if they had prior experiences of a child with JIA (moreso with a ‘protracted pathway’) or exposure to paediatric rheumatology in their training. Conversely JIA was more likely to be overlooked if the child had comorbidity such as learning disability or a chronic illness. Conclusions Care pathways are often ‘turbulent’ prior to a diagnosis of JIA with physical and emotional distress for families. There is need for greater awareness about JIA amongst health care professionals and observations of change (from family and non-health care professionals such as teachers) are key to trigger referral for paediatric rheumatology opinion.

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“Asking Too Much?”: Randomized N-of-1 Trial Exploring Patient Preferences and Measurement Reactivity to Frequent Use of Remote Multidimensional Pain Assessments in Children and Young People With Juvenile Idiopathic Arthritis (Preprint)

10.2196/preprints.14503 ◽

2019 ◽

Author(s):

Rebecca Rachael Lee ◽

Stephanie Shoop-Worrall ◽

Amir Rashid ◽

Wendy Thomson ◽

Lis Cordingley

Keyword(s):

Juvenile Idiopathic Arthritis ◽

Young People ◽

Patient Preferences ◽

Pain Interference ◽

Time Sampling ◽

Sampling Strategies ◽

Children And Young People ◽

Measurement Reactivity ◽

Pain Reporting ◽

Frequent Assessment

BACKGROUND Remote monitoring of pain using multidimensional mobile health (mHealth) assessment tools is increasingly being adopted in research and care. This assessment method is valuable because it is challenging to capture pain histories, particularly in children and young people in diseases where pain patterns can be complex, such as juvenile idiopathic arthritis (JIA). With the growth of mHealth measures and more frequent assessment, it is important to explore patient preferences for the timing and frequency of administration of such tools and consider whether certain administrative patterns can directly impact on children’s pain experiences. OBJECTIVE This study aimed to explore the feasibility and influence (in terms of objective and subjective measurement reactivity) of several time sampling strategies in remote multidimensional pain reporting. METHODS An N-of-1 trial was conducted in a subset of children and young people with JIA and their parents recruited to a UK cohort study. Children were allocated to 1 of 4 groups. Each group followed a different schedule of completion of MPT for 8 consecutive weeks. Each schedule included 2 blocks, each comprising 4 different randomized time sampling strategies, with each strategy occurring once within each 4-week block. Children completed MPT according to time sampling strategies: once-a-day, twice-a-day, once-a-week, and as-and-when pain was experienced. Adherence to each strategy was calculated. Participants completed the Patient-Reported Outcomes Measurement Information System Pain Interference Scale at the end of each week to explore objective reactivity. Differences in pain interference scores between time sampling strategies were assessed graphically and using Friedman tests. Children and young people and their parents took part in a semistructured interview about their preferences for different time sampling strategies and to explore subjective reactivity. RESULTS A total of 14 children and young people (aged 7-16 years) and their parents participated. Adherence to pain reporting was higher in less intense time sampling strategies (once-a-week=63% [15/24]) compared with more intense time sampling strategies (twice-a-day=37.8% [127/336]). There were no statistically significant differences in pain interference scores between sampling strategies. Qualitative findings from interviews suggested that children preferred once-a-day (6/14, 43%) and as-and-when pain reporting (6/14, 43%). Creating routine was one of the most important factors for successful reporting, while still ensuring that comprehensive information about recent pain was captured. CONCLUSIONS Once-a-day pain reporting provides rich contextual information. Although patients were less adherent to this preferred sampling strategy, once-a-day reporting still provides more frequent assessment opportunities compared with other less intense or overburdensome schedules. Important issues for the design of studies and care incorporating momentary assessment techniques were identified. We demonstrate that patient reporting preferences are key to accommodate and are important where data capture quality is key. Our findings support frequent administration of such tools, using daily reporting methods where possible.

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P28 Do we meet the needs of patients with non-inflammatory conditions referred to paediatric rheumatology?

Rheumatology ◽

10.1093/rheumatology/kez414.009 ◽

2019 ◽

Vol 58 (Supplement_4) ◽

Author(s):

Sofi Berrisford ◽

Isabel Carter ◽

Valentina Leone ◽

Marek Bohm

Keyword(s):

Pain Management ◽

Young People ◽

Occupational Therapy ◽

Conflicts Of Interest ◽

Pain Syndrome ◽

Holistic Approach ◽

Paediatric Rheumatology ◽

Initial Assessment ◽

Children And Young People ◽

Inflammatory Conditions

Poster presentation Tuesday 8 October Background Over one in four children and young people (CYP) present to paediatric rheumatology services with non-inflammatory conditions. However, there is a lack of evidence-based treatments and clinical guidelines supporting the management of these patients. Our project aims to determine whether CYP diagnosed with different non-inflammatory conditions and their families are satisfied with the service offered by the paediatric rheumatology department at Leeds General Infirmary. In addition, we wanted to assess which interventions they found most helpful and to check if there were differences between children and young people diagnosed with different non-inflammatory conditions. Methods 632 patients seen by Leeds paediatric rheumatology between July 2017 and June 2018 were diagnosed with non-inflammatory conditions; the three most common groups of conditions were symptomatic hypermobility (SH), chronic pain syndrome (CPS) and muscular back pain (MBP). We undertook a patient satisfaction survey, including patients reported assessment of their physical and psycho-social outcomes, focussing on these three groups only. 198 participants (80 SH; 74 CPS; 44 MBP) were invited to the study by sending them a postal questionnaire with self-addressed and stamped envelopes to return them. The questionnaire did not include any identifiable patient information but a different coloured paper was sent to patients with the three different groups of diagnoses to allow comparison of these groups. All had attended the paediatric rheumatology clinic for their initial assessment and had been referred on to appropriate management services as for the treating clinician including physiotherapy, occupational therapy, podiatry/orthotics, pain management, and/or psychology. Results A total of 33 filled questionnaires were received over the next 2 months including 4 (9.09%) from patients diagnosed with MBP, 11 (13.75%) from patients diagnosed with SH and 18 (24.32%) from patients diagnosed with CPS. Mann-Whitney-U calculations were performed to compare groups. The CPS patient group derived less benefit from physical therapies compared with non-CPS patients (U = 35.5, p = 0.0251) and that their reported mental health is worse than non-CPS patients (U = 31.5, p = 0.034). Within the CPS group, patients benefitted more from occupational therapy (U = 13.5, p = 0.01242) and pain management clinic (U = 9.5, p = 0.0226) than podiatry/orthotics services. When asked to rate their overall satisfaction out of 10, the median scores for the SH and MBP groups were 9 and 10 respectively, the median score for the CPS group was 5. Conclusion Our data would suggest that patients suffering with CPS would benefit from a more holistic approach including referrals to a psychologist, occupational therapy and the pain management team. The CPS group seemed most dissatisfied with the services provided by the clinic. The results of the project were fed back to the clinical team and we hope to repeat the survey in future after potential changes suggested by the survey have been implemented. Conflicts of Interest The authors declare no conflicts of interest.

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