A multidisciplinary approach to the assessment and management of disease-related foot impairments in juvenile idiopathic arthritis

Introduction: Juvenile idiopathic arthritis is a chronic inflammatory arthritis of childhood which often results in chronic, persistent and disabling foot impairments. Modern day clinical guidelines recommend early and aggressive therapeutic intervention in order to arrest inflammatory disease activity and preserve function. For these reasons, this thesis focuses on the development and evaluation of a contemporary new foot care programme. The programme of care comprises rapid access to targeted, individualised therapies informed by a sensitive imaging technique, and delivered by a multi disciplinary team. This is a logical and advanced extension of previous research within the field of paediatric rheumatology. Aims: The primary aim of the present work was to investigate the key methodological and procedural issues associated with evaluating a new foot care intervention under trial conditions for patients with JIA. Secondary aims were to estimate the levels of agreement between clinical and ultrasound examinations of foot disease, and the prevalence of sub- clinical foot disease in JIA. A discrete choice experiment questionnaire capable of eliciting and quantifying preferences for foot care was to be developed. The clinical and cost- effectiveness of this newly developed foot care programme were also explored. Methods: An experimental foot care programme was developed according to current evidence of best practice and expert opinion. An exploratory phase II non-pharmacological randomised-controlled trial and a full economic analysis were conducted in order to compare the experimental intervention with the current 'usual care' alternative. Patients with a definitive diagnosis of JIA and a history of foot and ankle arthritis were recruited from a paediatric rheumatology hospital outpatient clinic and randomised to 12 months of care in either treatment arm. At trial baseline, clinical and ultrasound examinations data from a pre- determined subset of participants were compared to estimate levels of agreement and subclinical disease. The primary outcome was change to the levels of foot-related impairments and disability which were assessed following 6 and 12 months of exposure to the interventions. A discrete choice experiment designed according to published design efficiency criteria was administered at baseline to determine parental preferences and willingness -to-pay for foot care. Costs of both interventions were estimated, and costs in relation to changes in foot function and quality of life outcomes were compared. Results: Of 126 patients identified, 85 were eligible, 44 were recruited, and 41 completed the study. 21 participants were allocated to receive the experimental intervention while 23 participants were allocated to receive 'usual care'. 3 participants in the 'usual care' arm self- withdrew prior to final follow up. The present work demonstrated that further phase I modelling and pre-clinical research is required prior to the implementation of a phase III definitive trial to evaluate similar experimental interventions. Multidisciplinary foot care appeared to be no more effective than 'usual care' for improving levels of foot impairments and disability. In terms of foot care service provision for children with HA, parents appear to prefer improvements in health outcomes over non-health outcomes and service process attributes. The experimental intervention was more costly over a 12 month period and thus non-cost-effective relative to the alternative intervention. Ultrasound and clinical examinations of foot disease were frequently discordant, and subclinical foot disease appears common in JIA. Conclusions: The primary fmdings from this thesis highlight areas requiring development through feasibility studies prior to the implementation of defmitive trials of multidisciplinary foot care. Secondary fmdings suggest that there were no sustainable improvements in functional outcomes following a programme of multidisciplinary foot care received by patients with established disease. Persistent levels of foot impairments and disability were exhibited despite targeted interventions. It is recommended that earlier intervention of this kind should be implemented in order to improve outcomes. The findings of this project provide an important foundation for future research into the optimisation of foot care for children and adolescents with JIA.EThOS - Electronic Theses Online ServiceGBUnited Kingdo