BackgroundChildhood onset uveitis comprises a group of rare inflammatory disorders characterized by clinical heterogeneity, chronicity, and uncertainties around long term outcomes. Standardized, detailed datasets with harmonized clinical definitions and terminology are needed to enable the clinical research necessary to stratify disease phenotype and interrogate the putative determinants of health outcomes. We aimed to develop a core routine clinical collection dataset for clinicians managing children with uveitis, suitable for multicenter and national clinical and experimental research initiatives.MethodsDevelopment of the dataset was undertaken in three phases: phase 1, a rapid review of published datasets used in clinical research studies; phase 2, a scoping review of disease or drug registries, national cohort studies and core outcome sets; and phase 3, a survey of members of a multicenter clinical network of specialists. Phases 1 and 2 provided candidates for a long list of variables for the dataset. In Phase 3, members of the UK's national network of stakeholder clinicians who manage childhood uveitis (the Pediatric Ocular Inflammation Group) were invited to select from this long-list their essential items for the core clinical dataset, to identify any omissions, and to support or revise the clinical definitions. Variables which met a threshold of at least 95% agreement were selected for inclusion in the core clinical dataset.ResultsThe reviews identified 42 relevant studies, and 9 disease or drug registries. In total, 138 discrete items were identified as candidates for the long-list. Of the 41 specialists invited to take part in the survey, 31 responded (response rate 78%). The survey resulted in inclusion of 89 data items within the final core dataset: 81 items to be collected at the first visit, and 64 items at follow up visits.DiscussionWe report development of a novel consensus core clinical dataset for the routine collection of clinical data for children diagnosed with non-infectious uveitis. The development of the dataset will provide a standardized approach to data capture able to support observational clinical studies embedded within routine clinical care and electronic patient record capture. It will be validated through a national prospective cohort study, the Uveitis in childhood prospective national cohort study (UNICORNS).
Background: Childhood uveitis is a rare inflammatory eye disease which is typically chronic, relapsing-remitting in nature, with an uncertain aetiology (idiopathic). Visual loss occurs due to structural damage caused by uncontrolled inflammation. Understanding of the determinants of long term outcome is lacking, including the predictors of therapeutic response or how to define disease control. Aims: To describe disease natural history and outcomes amongst a nationally representative group of children with non-infectious uveitis, describe the impact of disease course on quality of life for both child and family, and identify determinants of adverse visual, structural and developmental outcomes. Methods: UNICORNS is a prospective longitudinal multicentre cohort study of children newly diagnosed with uveitis about whom a core minimum clinical dataset will be collected systematically. Participants and their families will also complete patient-reported outcome measures annually from recruitment. The association of patient (child- and treatment- dependent) characteristics with outcome will be investigated using logistic and ordinal regression models which incorporate adjustment for within-child correspondence between eyes for those with bilateral disease and repeated outcomes measurement. Discussion: Through this population based, prospective longitudinal study of childhood uveitis, we will describe the characteristics of childhood onset disease. Early (1-2 years following diagnosis) outcomes will be described in the first instance, and through the creation of a national inception cohort, longer term studies will be enabled of outcome for affected children and families.
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Background/Objectives: There is a paucity of online educational content targeting children and young people with uveitis. We evaluated the impact of a co-designed patient education video on subjective and objective understanding of childhood uveitis. Subjects/Methods: Co-designed patient education media were produced in collaboration with the Childhood Uveitis Studies steering group and the Great Ormond Street Hospital Generation R Young People’s Advisory Group, and narrated by children. Patients managed within the Uveitis service at GOSH were invited to take part in a pre-post survey, undertaken immediately prior to and following viewing of a patient education video. Results: Forty-three patients participated. These were stratified according to age, duration of disease, and treatment type for analysis. Self-rated knowledge improved across all groups (p=0.001), particularly in those with a new diagnosis of uveitis (Z= -8.124, p <0.001). Objective knowledge scores improved across all questions, especially in younger children, those with new disease, and those on steroid only treatment (Z= -3.847, p <0.001, Z= -3.975, p <0.001, Z= -3.448, p <0.001; respectively). Most participants reported the videos to be easy to understand and with the right amount of information. All stated that they learned something new. Conclusions: Patient understanding of disease and treatment is crucial to achieving the best possible outcomes for this chronic, relapsing remitting and potentially blinding disorder. Our findings show the potential value of co-designed patient information videos, particularly benefitting younger patients and those with recently diagnosed disease as in our study. We suggest that other clinical teams could collaborate fruitfully with patient groups to develop similar videos and target possible misinformation, to potentially improve patient outcomes.
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