Transfer and transition of adolescents and young adults with juvenile idiopathic arthritis

Children with chronic conditions often associated with important and progressive morbidity can now live long enough to become adults with a manageable chronic disease. Expert lifetime care should be provided to these patients to maximise lifelong functioning and potential. Transition programmes are developed to prepare adolescents with chronic diseases to transfer to adult-centred care and to help them taking responsibility fortheir life and health. To date, no experimental studies examining the efficacy or effectiveness of a transition program were published. Thismay be due to the fact that research on this topic is methodologically challenging and require particular methodological approaches. Transitionprograms can be seen as complex interventions, needing research methods suitable for such interventions. Furthermore, transition programs are generally time-consuming, which limits its implementability. Therefore, it is worthwhile to develop brief transition programs that may have a higher likelihood to be implemented. Transition programs for persons with juvenile idiopathic arthritis JIA is a chronic inflammatory paediatric disorder, often resulting in short-term and long-term disability. The prevalence of JIA is estimated to range from 1 to 4 per 1000 children under the age of 16. For Belgium, it is estimated that there are up to 10000 children and adolescents with JIA. JIA has to be considered as a chronic condition that may persist in adulthood. Therefore, JIA forms an appropriate use case for studies on the effectiveness of transition programs. Empirical evidence on transition programs for young persons with JIA is predominantly conducted in theUK. Hence, generizability of these results to other health care systems is rather limited. The DON T RETARD project In order to develop and test a transition program for young people with JIA, the DON T RETARD project (D evices for the O ptimizatioN of TR ansfE r and T ransition of A dolescents with R heumatic D isorders) was established. The main objective of this dissertation, and thus also of the DON T RETARD project, is to explore the impact of a transition program implemented as a brief intervention in a paediatric rheumatology setting. In this dissertation, we used the Medical Research Council (MRC) framework to guide us in the development and evaluation of our transition programme . The original model of the MRC comprised an investigative sequence of five phases. For thefirst phase, the theoretical phase, published literature on the contentand effects of transition programs were scrutinized. In the second phase, the modelling phase, more understanding of the intervention s key components and its possible effects were developed. Four studies of the modelling phase were presented in this dissertation. After these preparatory studies, an exploratory trial was initiated. Inthe DON T RETARD project, a transition program for young people with JIA is designed as a brief intervention, and its clinical impact is evaluated. Studies in modelling phase For the modelling phase, it was important to have a good understanding how rheumatology practitioners look upon transfer and transition (chapter 2); where patients are cared for when they have reached adulthood (chapter 3); and how patients experienced to grow up with a rheumatic disorder (chapter 4) and to be transferred to adult care (chapter 5). Before a sound transition program could be developed, these issues were investigated in preparatory studies. Three main results were obtained. First, a proper transfer from paediatric to adult-focused rheumatology care is essential. Indeed, the majority of the rheumatology practitioners agreed that patients with an active rheumatic disorder should transfer to an adult rheumatologist so that they could continue with specialized care. Furthermore, we found that a structured transfer to adult rheumatology would have been more appropriate for patients who were no longer in follow-up. Indeed, although JIA patients with persistent disease and associated functional disabilities tend to remain in the rheumatology circuit, one-third of JIA patients older than 16 was no longer in follow-up. The majorityof these patients were in remission so one could judge that follow-up may be unnecessary. Yet, 16.7% of these patients had mild disabilities and 41.7% perceived mild pain. Further medical surveillance would probably benefit these patients. Second, the qualitative study showed that the physical impact of JIA, as well as its impact on medication, relationships and family, friends and perceptions was important. The physical impact of JIA involved functional limitations, pain, and fatigue. Taking medication properly was difficult; side effects were seen as a problem. With regard to relationships and family, JIA affected the subjects in their roles as family members and affected intimate relationships, pregnancy, and raising children. In fact, the majority of the patients were afraid to become pregnant or to have children. Some patients were afraid of what the future would bring. These results made clear that JIA has a significant impact on adolescents psychosocial health and that transitionalcare should incorporate specific components to improve the psychosocialhealth of these patients. Third, in the experiences and expectations ofadolescents with JIA who were transferred from paediatrics to adult-oriented healthcare, preparation to transfer , parental involvement and an adapted setting for late-adolescent or early adult were identified as three main themes. Young people with JIA prefer to have a say in the moment of transfer and in the reduction of parental involvement. The majority of the participants like their parents presence at the first consultation at the adult rheumatology department. They expect a healthcare setting adapted to their needs and the possibility to meet peers in this setting. These experiences and expectations of our participating adolescents with JIA helped us to configure the design of the transition program. The studies that were conducted in the modelling phase allowed us to identify specific key components that ought to form the basis for potentially feasible transition programs. Eight key components of the transition program implemented in the DON T RETARDproject were identified: (a) a transition coordinator; ( b) providinginformation and education about JIA and medication management, health behaviour, dealing with fatigue, school, friends and any problems with medication adherence ; ( c) availability by telephone; (d) informationabout and contact with the adult rheumatology program ; ( e) guidance of parents ; ( f) meeting with peers ; ( g) a transfer plan ; (h) t he actual transfer to adult rheumatology programme . Clinical impact of a brief transition program for young people with JIA As part of the exploratory phase of the MRC framework, the next step in the DON T RETARD project was to perform a study in which the newly developed transition program was tested. We presented the content of the transition program for young people with JIA devised as a brief intervention, and described the rationale and design of the evaluation of this transition program, using a mixed methods approach.Further, the clinical impact of our transition program was investigated. The impact of the transition program was evaluated in a quantitative study using an embedded experimental design . As primary outcome,an improved physical, psychosocial, and rheumatic-specific health status, was detected. With respect to the secondary outcomes, an improved quality of life and optimized parenting behaviours were observed. This brief transition program met to a large extent the needs of thepatients as expressed in the modelling studies. The provided psychosocial guidance by the transition coordinator (TC) felt to be pivotal. The adolescent information day may also have had a substantial contribution to their psychosocial well-being and feelings of reassurance. Furthermore, the adolescents expected a shift in the parental role. From the qualitative study, we learned that especially the gradual approach of handing over the responsibility in health behaviour and disease management to their child was appreciated by the majority of the parents. The clear message of the TC and her companionship in the transition process encouraged the parents to reduce their supervisory role. Sharing their parental concerns, anxieties, and also thankfulness with other parents, felt as a relief. Conversely, the intervention didnot have the expected positive impact on medication adherence. More anticipatory guidance discussions to be included in our transition program could meet the specific adherence-enhancing needs. Next, illness-relatedknowledge in patients only increased a little over time, as shown in the longitudinal analyses. However, the knowledge of patients who receivedthe intervention was not better than that of controls who did not go through a transition program. Alternative education techniques, other thanproviding brochures or referring to websites, are needed. Concerning fatigue, we observed no difference or slightly more fatigue occurring overtime, but less fatigue when compared with the control group. We concluded that proper assessment of and recommendations in dealing with fatigueshould be an indispensable element in individual guidance offered by the TC. Since the design of this study not permit us to draw conclusions in terms of effectiveness of the transition program, a definitive trial using an appropriate design should be conducted. A randomized controlled trial is required to draw firm conclusions about the effectiveness of the transition program. Concerning long term implementation research, continuous evaluation and SWOT-analysis could be recommended. Finally, increasing collaboration among paediatric and internal specialties in the wide array of chronic childhood-conditions is essential to develop an appropriate and disease-specific transition program to all adolescents with chronic disorders.status: publishe