Proposal for a definition for response to treatment, inactive disease and damage for JIA associated uveitis based on the validation of a uveitis related JIA outcome measures from the Multinational Interdisciplinary Working Group for Uveitis in Childhood (MIWGUC)
Por:
Foeldvari I, Klotsche J, Simonini G, Edelsten C, Angeles-Han ST, Bangsgaard R, de Boer J, Brumm G, Torrent RB, Constantin T, DeLibero C, Diaz J, Gerloni VM, Guedes M, Heiligenhaus A, Kotaniemi K, Leinonen S, Minden K, Miranda V, Miserocchi E, Nielsen S, Niewerth M, Pontikaki I, García de Vicuña-Muñoz C, Zilhao C, Yeh S, Anton-Lopez J and Calzada-Hernández J
Publicada:
1 oct 2019
Ahead of Print:
1 oct 2019
Resumen:
Background JIA-associated uveitis (JIAU) is a serious, sight-threatening disease with significant long-term complications and risk of blindness, even with improved contemporary treatments. The MIWGUC was set up in order to propose specific JIAU activity and response items and to validate their applicability for clinical outcome studies. Methods The group consists of 8 paediatric rheumatologists and 7 ophthalmologists. A consensus meeting took place on November 2015 in Barcelona (Spain) with the objective of validating the previously proposed measures. The validation process was based on the results of a prospective open, international, multi-centre, cohort study designed to validate the outcome measures proposed by the initial MIWGUC group meeting in 2012. The meeting used the same Delphi and nominal group technique as previously described in the first paper from the MIWGUC group (Arthritis Care Res 64:1365-72, 2012). Patients were included with a diagnosis of JIA, aged less than 18 years, and with active uveitis or an uveitis flare which required treatment with a disease-modifying anti-rheumatic drug. The proposed outcome measures for uveitis were collected by an ophthalmologist and for arthritis by a paediatric rheumatologist. Patient reported outcome measures were also measured. Results A total of 82 patients were enrolled into the validation cohort. Fifty four percent (n = 44) had persistent oligoarthritis followed by rheumatoid factor negative polyarthritis (n = 15, 18%). The mean uveitis disease duration was 3.3 years (SD 3.0). Bilateral eye involvement was reported in 65 (79.3%) patients. The main findings are that the most significant changes, from baseline to 6 months, are found in the AC activity measures of cells and flare. These measures correlate with the presence of pre-existing structural complications and this has implications for the reporting of trials using a single measure as a primary outcome. We also found that visual analogue scales of disease activity showed significant change when reported by the ophthalmologist, rheumatologist and families. The measures formed three relatively distinct groups. The first group of measures comprised uveitis activity, ocular damage and the ophthalmologists' VAS. The second comprised patient reported outcomes including disruption to school attendance. The third group consisted of the rheumatologists' VAS and the joint score. Conclusions We propose distinctive and clinically significant measures of disease activity, severity and damage for JIAU. This effort is the initial step for developing a comprehensive outcome measures for JIAU, which incorporates the perspectives of rheumatologists, ophthalmologists, patients and families.
Filiaciones:
Foeldvari I:
Head of the Hamburg Centre for Pediatric and Adolescence Rheumatology Centre for Treatment of Scleroderma and Uveitis in Childhood and Adolescence Teaching Unit of the Asklepios Campus of the Semmelweis Medical School, Budapest An der Schön Klinik Hamburg Eilbek Dehnhaide, 120 22081, Hamburg, Germany.
Klotsche J:
German Rheumatism Research Centre, 10117, Berlin, Germany
Institute for Social Medicine, Epidemiology, and Health Economics, Charité Universitaetsmedizin Berlin, Berlin, Germany
Simonini G:
Rheumatology Unit- A. Meyer Children's Hospital- NEUROFARBA Department, University of Florence, Florence, Italy
Edelsten C:
Dept Rheumatology, Great Ormond Street Hospital, Great Ormond Street, London, UK
Angeles-Han ST:
Division of Rheumatology, Cincinnati Children's Hospital Medical Center, 3333 Burnett Avenue, Cincinnati, OH 45229
Department of Pediatrics, University of Cincinnati, Cincinnati, OH, USA
Bangsgaard R:
Department of Ophthalmology, Copenhagen University Hospital Glostrup/Rigshospitalet, Copenhagen, Denmark
de Boer J:
UMC Utrecht, Utrecht, Netherlands
Brumm G:
Universitätsklinikum Hamburg-Eppendorf, Hamburg, Germany
Torrent RB:
Pediatric Rheumatology Department, Hospital Sant Joan de Déu, Barcelona, Spain
Institut de Recerca Sant Joan de Déu, Barcelona, Spain
Constantin T:
2nd Department of Pediatrics, Semmelweis University, Budapest, Hungary
DeLibero C:
AOU Meyer, Florence, Italy
Diaz J:
Ophtalmology Department, Hospital Sant Pau, Barcelona, Spain
Ophtalmology Department, Hospital Sant Joan de Déu, Barcelona, Spain
Gerloni VM:
Università di Milano - Istituto Gaetano Pini, Milan, Italy
Guedes M:
Pediatric Rheumatology Unit, Centro Hospitalar Universitário do Porto, Porto, Portugal
Heiligenhaus A:
St. Franziskus-Hospital Münster, Muenster, Germany
Kotaniemi K:
Department of Ophthalmology, Helsinki University Hospital, Helsinki, Finland
Leinonen S:
Department of Ophthalmology, Helsinki University Hospital, Helsinki, Finland
Minden K:
German Rheumatism Research Centre, 10117, Berlin, Germany
Charité - Universitätsmedizin Berlin, corporate member of Freie Universität Berlin, Humboldt-Universität zu Berlin, and Berlin Institute of Health, Department of Rheumatology and Clinical Immunology, Berlin, Germany
Miranda V:
Pediatric Ophthalmologist at the Centro Hospitalar Universitario do Porto, Teaching Unit of the Abel Salazar Institute of Biomedical Sciences, Largo do Prof. Abel Salazar, 4099-001, Porto, Portugal
Miserocchi E:
Ocular Immunology and Uveitis Service, Department of Ophthalmology, San Raffaele Scientific Institute, Via Olgettina 60, 20122, Milan, Italy
Nielsen S:
Department of Ophthalmology, Copenhagen University Hospital Glostrup/Rigshospitalet, Copenhagen, Denmark
Niewerth M:
German Rheumatism Research Centre, 10117, Berlin, Germany
Pontikaki I:
Università di Milano - Istituto Gaetano Pini, Milan, Italy
García de Vicuña-Muñoz C:
Pediatric Rheumatology Department, Hospital Sant Joan de Déu, Barcelona, Spain
Zilhao C:
Pediatric Rheumatology Unit, Centro Hospitalar Universitário do Porto, Porto, Portugal
Yeh S:
Emory Clinic, Atlanta, USA
Anton-Lopez J:
Pediatric Rheumatology Department, Hospital Sant Joan de Déu, Barcelona, Spain
Institut de Recerca Sant Joan de Déu, Barcelona, Spain
Department of Surgery and Surgery Specializations. Universitat de Barcelona, Barcelona, Spain
Calzada-Hernández J:
Pediatric Rheumatology Department, Hospital Sant Joan de Déu, Barcelona, Spain
Institut de Recerca Sant Joan de Déu, Barcelona, Spain
Green Submitted, gold
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