33 results on '"Saurenmann RK"'
Search Results
2. Maximal inter-incisal distance in healthy school children in Switzerland
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Saurenmann RK, Kellenberger CJ, Mueller L, Langerweger C, and van Waes H
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Pediatrics ,RJ1-570 ,Diseases of the musculoskeletal system ,RC925-935 - Published
- 2011
- Full Text
- View/download PDF
3. Are growing pains a parasomnia?
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Jenni O, Werner H, Aeschlimann F, and Saurenmann RK
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Pediatrics ,RJ1-570 ,Diseases of the musculoskeletal system ,RC925-935 - Published
- 2011
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4. Observation of juvenile idiopathic arthritis in children with MEVF gene mutations in Armenia
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Gayane Khloyan, Saurenmann RK, Gayane Amaryan, and Alan Karibian
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Pediatrics ,RJ1-570 ,Diseases of the musculoskeletal system ,RC925-935 - Published
- 2008
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5. Juvenile Psoriatic Arthritis (JPsA) clinical features and outcome of 119 patients
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Aviel Y Butbul, Tyrrell PN, Feldman BM, Laxer RM, Saurenmann RK, Spiegel L, Cameron B, Tse S, and Silverman ED
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Pediatrics ,RJ1-570 ,Diseases of the musculoskeletal system ,RC925-935 - Published
- 2008
- Full Text
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6. New and updated recommendations for the treatment of juvenile idiopathic arthritis associated uveitis and idiopathic chronic anterior uveitis
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Foeldvari I, Maccora I, Petrushkin H, Rahman N, Anton-Lopez J, de Boer J, Calzada J, Carreras E, Diaz J, Edelsten C, Angeles-Han ST, Heiligenhaus A, Miserocchi E, Nielsen S, Saurenmann RK, Stuebiger N, Baquet-Walscheid K, Furst D, and Simonini G
- Abstract
OBJECTIVE: The Multinational Interdisciplinary Working Group for Uveitis in Childhood (MIWGUC) identified the need to update the current guidelines and worked to produce this document, which should serve to guide clinicians managing children with juvenile idiopathic arthritis associated uveitis (JIAU) and idiopathic chronic anterior uveitis (CAU). METHODS: The group analysed the literature published between December 2014- June 2020 after a systematic literature review conducted by two clinicians. Paediatric rheumatologists were paired with ophthalmologists to review the eligible 37 publications. The search criteria were selected to reflect those used for the 2018 SHARE recommendations in order to provide an update, rather than a replacement for that publication. The consensus meeting was moderated by Prof Dan Furst MD, UCLA, Los Angeles, USA. The summary of the current evidence for each SHARE recommendation was presented to the expert committee. These were then discussed and revised during a video consensus meeting on the 22(nd) of January 2021 with 14 voting participants, using a Nominal Group Technique to reach consensus. RESULTS: JIAU treatment was extended to include CAU. Fourteen recommendations regarding treatment of JIAU und CAU with > 80% agreement were accepted. CONCLUSION: An update to the previous 2018 SHARE recommendations for the treatment of children with JIAU with the addition of CAU was created using an evidence-based consensus process. This guideline should help support clinicians to care for children and young people with chronic anterior uveitis.
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- 2022
7. Phenotypic variability and disparities in treatment and outcomes of childhood arthritis throughout the world: an observational cohort study
- Author
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Consolaro, A, Giancane, G, Alongi, A, van Dijkhuizen, Ehp, Aggarwal, A, Al-Mayouf, Sm, Bovis, F, De Inocencio, J, Demirkaya, E, Flato, B, Foell, D, Garay, Sm, Lazăr, C, Lovell, Dj, Montobbio, C, Miettunen, P, Mihaylova, D, Nielsen, S, Orban, I, Rumba-Rozenfelde, I, Magalhães, C, Shafaie, N, Susic, G, Trachana, M, Wulffraat, N, Pistorio, A, Martini, A, Ruperto, N, Ravelli, A &, Abdwani R, Aghighi, Y, Aiche, Mf, Ailioaie, C, Aktay Ayaz, N, Al-Abrawi, S, Alexeeva, E, Anton, J, Apostol, A, Arguedas, O, Avcin, T, Barone, P, Berntson, L, Boteanu, Al, Boyko, Y, Burgos-Vargas, R, Calvo Penades, I, Chédeville, G, Cimaz, R, Civino, A, Consolini, R, Constantin, T, Cuttica, R, Dallos, T, Martin, N, Magni-Manzoni, S, De Cunto, C, Dolezalova, P, Ekelund, M, El Miedany, Y, Espada, G, Estmann Christensen, A, Foeldvari, I, Gallizzi, R, Ganser, G, Gerloni, V, Haas, Jp, Harel, L, Harjacek, M, Hashad, S, Herlin, T, Herrera, C, Hofer, M, Holzinger, D, Horneff, G, Huppertz, Hi, Iagăru, N, Ibanez Estrella, A, Ioseliani, M, Joos, R, Knupp Oliveira, S, Kamphuis, S, Kasapcopur, O, Katsicas, Mm, Khubchandani, R, Kondi, A, Kröger, L, La Torre, F, Laday, M, Lahdenne, P, Maggio, Mc, Magnolia, Mg, Malagon, C, Malin, M, Martino, S, Melo-Gomes, Ja, Mesa-Del-Castillo, P, Militaru, A, Minden, K, Miniaci, A, Moradinejad, Mh, Morel Ayala, Z, Nikishina, I, Norambuena, X, Nordal, Eb, Pagava, K, Panaviene, V, Pastore, S, Pieropan, S, Podda, Ra, Pruunsild, C, Putto-Laurila, A, Quartier, P, Remesal, A, Rigante, Donato, Ringold, S, Rutkowska-Sak, L, Rygg, M, Saurenmann, Rk, Sawhney, S, Scott, C, Shiari, R, Smolewska, E, Sozeri, B, Swart, Jf, Sztajnbok, F, Torcoletti, M, Tsitsami, E, Tzaribachev, N, Unsal, E, Uziel, Y, Vähäsalo, P, Varbanova, B, Vargova, V, Vesely, R, Vijatov-Djuric, G, Vilaiyuk, S, Vojinovic, J, Vougiouka, O, Weiss, P, Wouters, C, Rigante D (ORCID:0000-0001-7032-7779), Consolaro, A, Giancane, G, Alongi, A, van Dijkhuizen, Ehp, Aggarwal, A, Al-Mayouf, Sm, Bovis, F, De Inocencio, J, Demirkaya, E, Flato, B, Foell, D, Garay, Sm, Lazăr, C, Lovell, Dj, Montobbio, C, Miettunen, P, Mihaylova, D, Nielsen, S, Orban, I, Rumba-Rozenfelde, I, Magalhães, C, Shafaie, N, Susic, G, Trachana, M, Wulffraat, N, Pistorio, A, Martini, A, Ruperto, N, Ravelli, A &, Abdwani R, Aghighi, Y, Aiche, Mf, Ailioaie, C, Aktay Ayaz, N, Al-Abrawi, S, Alexeeva, E, Anton, J, Apostol, A, Arguedas, O, Avcin, T, Barone, P, Berntson, L, Boteanu, Al, Boyko, Y, Burgos-Vargas, R, Calvo Penades, I, Chédeville, G, Cimaz, R, Civino, A, Consolini, R, Constantin, T, Cuttica, R, Dallos, T, Martin, N, Magni-Manzoni, S, De Cunto, C, Dolezalova, P, Ekelund, M, El Miedany, Y, Espada, G, Estmann Christensen, A, Foeldvari, I, Gallizzi, R, Ganser, G, Gerloni, V, Haas, Jp, Harel, L, Harjacek, M, Hashad, S, Herlin, T, Herrera, C, Hofer, M, Holzinger, D, Horneff, G, Huppertz, Hi, Iagăru, N, Ibanez Estrella, A, Ioseliani, M, Joos, R, Knupp Oliveira, S, Kamphuis, S, Kasapcopur, O, Katsicas, Mm, Khubchandani, R, Kondi, A, Kröger, L, La Torre, F, Laday, M, Lahdenne, P, Maggio, Mc, Magnolia, Mg, Malagon, C, Malin, M, Martino, S, Melo-Gomes, Ja, Mesa-Del-Castillo, P, Militaru, A, Minden, K, Miniaci, A, Moradinejad, Mh, Morel Ayala, Z, Nikishina, I, Norambuena, X, Nordal, Eb, Pagava, K, Panaviene, V, Pastore, S, Pieropan, S, Podda, Ra, Pruunsild, C, Putto-Laurila, A, Quartier, P, Remesal, A, Rigante, Donato, Ringold, S, Rutkowska-Sak, L, Rygg, M, Saurenmann, Rk, Sawhney, S, Scott, C, Shiari, R, Smolewska, E, Sozeri, B, Swart, Jf, Sztajnbok, F, Torcoletti, M, Tsitsami, E, Tzaribachev, N, Unsal, E, Uziel, Y, Vähäsalo, P, Varbanova, B, Vargova, V, Vesely, R, Vijatov-Djuric, G, Vilaiyuk, S, Vojinovic, J, Vougiouka, O, Weiss, P, Wouters, C, and Rigante D (ORCID:0000-0001-7032-7779)
- Abstract
BACKGROUND: To our knowledge, the characteristics and burden of childhood arthritis have never been studied on a worldwide basis. We aimed to investigate, with a cross-sectional study, the prevalence of disease categories, treatment methods, and disease status in patients from across different geographical areas and from countries with diverse wealth status. METHODS: In this multinational, cross-sectional, observational cohort study, we asked international paediatric rheumatologists from specialised centres to enrol children with a diagnosis of juvenile idiopathic arthritis, according to International League of Associations for Rheumatology criteria, who were seen consecutively for a period of 6 months. Each patient underwent retrospective and cross-sectional assessments, including measures of disease activity and damage and questionnaires on the wellbeing and quality of life of the children. We qualitatively compared the collected data across eight geographical areas, and we explored an association between disease activity and damage and a country's gross domestic product (GDP) with a multiple logistic regression analysis. FINDINGS: Between April 4, 2011, and Nov 21, 2016, 9081 patients were enrolled at 130 centres in 49 countries, grouped into eight geographical areas. Systemic arthritis (125 [33·0%] of 379 patients) and enthesitis-related arthritis (113 [29·8%] of 379) were more common in southeast Asia, whereas oligoarthritis was more prevalent in southern Europe (1360 [56·7%] of 2400) and rheumatoid factor-negative polyarthritis was more frequent in North America (165 [31·5%] of 523) than in the other areas. Prevalence of uveitis was highest in northern Europe (161 [19·1%] of 845 patients) and southern Europe (450 [18·8%] of 2400) and lowest in Latin America (54 [6·4%] of 849), Africa and Middle East (71 [5·9%] of 1209), and southeast Asia (19 [5·0%] of 379). Median age at disease onset was lower in southern Europe (3·5 years, IQR 1·9-7·3) than in other regions
- Published
- 2019
8. Toward Establishing a Standardized Magnetic Resonance Imaging Scoring System for Temporomandibular Joints in Juvenile Idiopathic Arthritis
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Tolend MA, Twilt M, Cron RQ, Tzaribachev N, Guleria S, von Kalle T, Koos B, Miller E, Stimec J, Vaid Y, Larheim TA, Herlin T, Spiegel L, Inarejos E, Moineddin R, van Rossum MA, Saurenmann RK, Doria AS, and Kellenberger CJ
- Subjects
musculoskeletal diseases - Abstract
OBJECTIVE: The temporomandibular joints (TMJs) are frequently affected in children with juvenile idiopathic arthritis (JIA). Early detection is challenging, as major variation is present in scoring TMJ pathology on magnetic resonance imaging (MRI). Consensus-driven development and validation of an MRI scoring system for TMJs has important clinical utility in timely improvement of diagnosis and serving as an outcome measure. We report on a multi-institutional collaboration toward developing a TMJ MRI scoring system for JIA. METHODS: Seven readers independently assessed MRI scans from 21 patients (42 TMJs, from patients ages 6-16 years) using 3 existing MRI scoring systems from American, German, and Swiss institutions. Reliability scores, scoring system definitions, and items were discussed among 10 JIA experts through 2 rounds of Delphi surveys, nominal group voting, and subsequent consensus meetings to create a novel TMJ MRI scoring system. RESULTS: Average-measure absolute agreement intraclass correlation coefficients (avICCs) for the total scores of all 3 scoring systems were highly reliable at 0.96 each. Osteochondral items showed higher reliability than inflammatory items. An additive system was deemed preferable for assessing minor joint changes over time. Eight items were considered sufficiently reliable and/or important for integration into the consensus scoring system: bone marrow edema and enhancement (avICC 0.57-0.61, smallest detectable difference [SDD] ± 45-63% prior to redefining), condylar flattening (avICC 0.95-0.96, SDD ± 23-28%), effusions (avICC 0.85-0.88, SDD ± 25-26%), erosions (avICC 0.94, SDD ± 20%), synovial enhancement and thickening (previously combined, avICC 0.90-0.91, SDD ± 33%), and disk abnormalities (avICC 0.90, SDD ± 19%). CONCLUSION: A novel TMJ MRI scoring system was developed by consensus. Further iterative refinements and reliability testing are warranted in upcoming studies.
- Published
- 2018
9. The provisional paediatric rheumatology international trials organisation/Americal college of rheumatology/European league against reumatism disease activity core set for the evaluation of response to therapy in juvenile dermatomyositis: a prospective validation study
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Ruperto, N, Ravelli, A, Pistorio, A, Ferriani, V, Calvo, I, Ganser, G, Brunner, J, Dannecker, G, Silva, CA, Stanevicha, V, ten Cate, R, Smit, LWA, Voygiyka, O, Fischbach, M, Foeldvari, I, Hilario, O, Modesto, C, Saurenmann, RK, Sauvain, MJ, Scheibel, I, Sommelet, D, Tambic-Bukovac, L, Barcellona, R, Brik, R, Ehl, S, Jovanovic, M, Rovensky, J, Bagnasco, F, Lovell, DJ, Martini, A, and Pediatrics
- Published
- 2008
10. Are growing pains a parasomnia?
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Aeschlimann, F, primary, Werner, H, additional, Jenni, O, additional, and Saurenmann, RK, additional
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- 2011
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11. Association of the CD226 (DNAM-1) Gly307Ser polymorphism with juvenile idiopathic arthritis
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Reinards, THCM, primary, Albers, HM, additional, Brinkman, DMC, additional, Kamphuis, SSM, additional, van Rossum, MAJ, additional, Hoppenreijs, EPAH, additional, Girschick, HJ, additional, Wouters, C, additional, Saurenmann, RK, additional, Houwing-Duistermaat, JJ, additional, Toes, REM, additional, Huizinga, TWJ, additional, ten Cate, R, additional, and Schilham, MW, additional
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- 2011
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12. Maximal inter-incisal distance in healthy school children in Switzerland
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van Waes, H, primary, Langerweger, C, additional, Mueller, L, additional, Kellenberger, CJ, additional, and Saurenmann, RK, additional
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- 2011
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13. Association of the CCR5 Δ32 variant with juvenile idiopathic arthritis in a meta-analysis
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Reinards, THCM, primary, Albers, HM, additional, Brinkman, DMC, additional, Kamphuis, SSM, additional, van Rossum, MAJ, additional, Hoppenreijs, EPAH, additional, Girschick, HJ, additional, Wouters, C, additional, Saurenmann, RK, additional, Houwing-Duistermaat, JJ, additional, Toes, REM, additional, Huizinga, TWJ, additional, ten Cate, R, additional, and Schilham, MW, additional
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- 2011
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14. Temporomandibular joint arthritis in patients with juvenile idiopathic arthritis: efficacy of intraarticular corticosteroid injection as measured by MRI and clinical examination
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Schroeder, S, primary, Cannizzaro, E, additional, Kellenberger, C, additional, Peltomäki, T, additional, and Saurenmann, RK, additional
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- 2008
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15. Association of the autoimmunity locus 4q27 with juvenile idiopathic arthritis.
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Albers HM, Kurreeman FA, Stoeken-Rijsbergen G, Brinkman DM, Kamphuis SS, van Rossum MA, Girschick HJ, Wouters C, Saurenmann RK, Hoppenreijs E, Slagboom P, Houwing-Duistermaat JJ, Verduijn W, Huizinga TW, Ten Cate R, Toes RE, and Schilham MW
- Abstract
OBJECTIVE: Juvenile idiopathic arthritis (JIA) is characterized by chronic arthritis and an autoimmune etiology. In several autoimmune diseases, including rheumatoid arthritis (RA), an association with the 4q27 locus has been reported. We undertook this study to investigate the possible role of the 4q27 locus in JIA. METHODS: A case-control association study was conducted, with a total of 655 Caucasian JIA patients and 791 healthy controls divided into 2 independent sample sets. The rs6822844 marker in the 4q27 locus was genotyped. RESULTS: In the first and larger sample set, a 5% decrease in T allele frequency was observed in patients compared with controls (allelic odds ratio [OR] 0.72 [95% confidence interval 0.55-0.95], P = 0.019), and in the second set, a 3% decrease was observed (allelic OR 0.81 [95% confidence interval 0.61-1.09], P = 0.169). The combined data set generated an OR of 0.76 (95% confidence interval 0.62-0.93, P = 7.08 x 10(-3)). When the different JIA subtypes were analyzed individually, significant decreases were seen in the subtypes with a polyarticular course of disease (extended oligoarthritis [P = 0.019] and rheumatoid factor-negative polyarthritis [P = 0.038]). CONCLUSION: Our findings suggest that the 4q27 locus, previously reported to be associated with RA, type 1 diabetes mellitus, celiac disease, and psoriatic arthritis, is also associated with susceptibility to JIA. [ABSTRACT FROM AUTHOR]
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- 2009
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16. Epidemiology of juvenile idiopathic arthritis in a multiethnic cohort: ethnicity as a risk factor.
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Saurenmann RK, Rose JB, Tyrrell P, Feldman BM, Laxer RM, Schneider R, and Silverman ED
- Abstract
OBJECTIVE: To study the influence of ethnicity on the risk of developing juvenile idiopathic arthritis (JIA) in a multiethnic community of patients with unrestricted access to health care. METHODS: A questionnaire on ethnicity was distributed to all patients with JIA being followed up at the Hospital for Sick Children in Toronto, Ontario, Canada. Of 1,082 patients, 859 (79.4%) responded to the questionnaire. To calculate the relative risk (RR) of developing JIA in this study cohort, the results were compared with data from the age-matched general population of the Toronto metropolitan area (TMA) as provided in the 2001 census from Statistics Canada. RESULTS: European descent was reported by 69.7% of the patients with JIA compared with a frequency of 54.7% in the TMA general population, whereas a statistically significantly lower than expected percentage of the patients with JIA reported having black, Asian, or Indian subcontinent origin. Children of European origin had a higher RR for developing any of the JIA subtypes except polyarticular rheumatoid factor (RF)-positive JIA, and were particularly more likely to develop the extended oligoarticular and psoriatic subtypes. A higher frequency of enthesitis-related JIA was observed among patients of Asian origin, while those of black origin or native North American origin were more likely to develop polyarticular RF-positive JIA. CONCLUSION: In this multiethnic cohort, European descent was associated with a significantly increased risk of developing JIA, and the distribution of JIA subtypes differed significantly across ethnic groups. [ABSTRACT FROM AUTHOR]
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- 2007
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17. Prevalence, risk factors, and outcome of uveitis in juvenile idiopathic arthritis: a long-term followup study.
- Author
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Saurenmann RK, Levin AV, Feldman BM, Rose JB, Laxer RM, Schneider R, and Silverman ED
- Abstract
OBJECTIVE: To assess the prevalence, risk factors, and long-term outcome of uveitis in patients with juvenile idiopathic arthritis (JIA). METHODS: An inception cohort of all 1,081 patients diagnosed as having JIA at a single tertiary care center was established. A questionnaire and followup telephone calls were used to confirm the diagnosis of uveitis. Ophthalmologists' records of patients with uveitis were collected. Kaplan-Meier and Cox regression analyses were used to assess risk factors for developing uveitis and for complications of uveitis. RESULTS: After a mean followup time of 6.9 years, 142 of 1,081 patients (13.1%) had developed uveitis. Risk factors were young age at diagnosis, female sex, antinuclear antibody positivity, and the subtype of JIA. The relative contribution of these risk factors was different for the different subtypes of JIA. Until the end of the study, uveitis complications had developed in 53 of 142 patients with uveitis (37.3%; 4.9% of the total cohort). Only 16 of 175 involved eyes (9.1%) in 14 of 108 patients (13%; 1.3% of the total cohort) for whom ophthalmology reports were available had best corrected visual acuity less than 20/40 (mean followup time for uveitis of 6.3 years). Abnormal vision was associated with synechiae or cataract. CONCLUSION: Risk factors for developing uveitis were different among subtypes of JIA. The long-term outcome of JIA-associated uveitis in our cohort was excellent despite the high rate of complications. [ABSTRACT FROM AUTHOR]
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- 2007
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18. Uncovering the hidden socioeconomic impact of juvenile idiopathic arthritis and paving the way for other rare childhood diseases: an international, cross-disciplinary, patient-centered approach (PAVE Consortium).
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Marshall DA, Gerber B, Currie GR, Antón J, De Somer L, Dey M, Egert T, Egert Y, Henan L, Klotsche J, Mifsut LM, Minden K, Normand C, Porte D, Saurenmann RK, Swart JF, Uziel Y, Wilson J, Wouters C, Ziv A, and Benseler SM
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- Humans, Child, Cost of Illness, Europe, Arthritis, Juvenile economics, Patient-Centered Care economics, Rare Diseases economics
- Abstract
Background: Juvenile idiopathic arthritis (JIA) refers to a heterogeneous group of rheumatic conditions in children. Novel drugs have greatly improved disease outcomes; however, outcomes are impacted by limited awareness of the importance of early diagnosis and adequate treatment, and by differences in access across health systems. As a result, patients with JIA continue to be at risk for short- and long-term morbidity, as well as impacts on virtually all aspects of life of the child and family., Main Body: Literature on the socioeconomic burden of JIA is largely focused on healthcare costs, and the impact of JIA on patients, families, and communities is not well understood. High quality evidence on the impact of JIA is needed to ensure that patients are receiving necessary support, timely diagnostics, and adequate treatment, and to inform decision making and resource allocation. This commentary introduces the European Joint Programme on Rare Diseases: Producing an Arthritis Value Framework with Economic Evidence: Paving the Way for Rare Childhood Diseases (PAVE) project, which will co-develop a patient-informed value framework to measure the impact of JIA on individuals and on society. With a patient-centered approach, fundamental to PAVE is the involvement of three patient advocacy organizations from Canada, Israel, and Europe, as active research partners co-designing all project phases and ensuring robust patient and family engagement. The framework will build on the findings of projects from six countries: Canada, Germany, Switzerland, Spain, Israel, and Belgium, exploring costs, outcomes (health, well-being), and unmet needs (uveitis, mental health, equity)., Conclusion: This unique international collaboration will combine evidence on costs (from family to societal), outcomes (clinical, patient and family outcomes), and unmet needs, to co-design and build a framework with patients and families to capture the full impact of JIA. The framework will support the development of high-quality evidence, encompassing economic and clinical considerations, unmet needs, and patient perspectives, to inform equitable resource allocation, health system planning, and quality of care better aligned with the needs of children with JIA, their families, and communities. Knowledge gained from this novel approach may pave the way forward to be applied more broadly to other rare childhood diseases., (© 2024. The Author(s).)
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- 2024
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19. New and Updated Recommendations for the Treatment of Juvenile Idiopathic Arthritis-Associated Uveitis and Idiopathic Chronic Anterior Uveitis.
- Author
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Foeldvari I, Maccora I, Petrushkin H, Rahman N, Anton J, de Boer J, Calzada-Hernández J, Carreras E, Diaz J, Edelsten C, Angeles-Han ST, Heiligenhaus A, Miserocchi E, Nielsen S, Saurenmann RK, Stuebiger N, Baquet-Walscheid K, Furst D, and Simonini G
- Subjects
- Child, Humans, Adolescent, Europe, Arthritis, Juvenile complications, Uveitis complications, Rheumatology, Uveitis, Anterior
- Abstract
Objective: The Multinational Interdisciplinary Working Group for Uveitis in Childhood identified the need to update the current guidelines, and the objective here was to produce this document to guide clinicians managing children with juvenile idiopathic arthritis-associated uveitis (JIAU) and idiopathic chronic anterior uveitis (CAU)., Methods: The group analyzed the literature published between December 2014 and June 2020 after a systematic literature review conducted by 2 clinicians. Pediatric rheumatologists were paired with ophthalmologists to review the eligible 37 publications. The search criteria were selected to reflect those used for the 2018 Single Hub and Access point for pediatric Rheumatology in Europe (SHARE) recommendations, in order to provide an update, rather than a replacement for that publication. The summary of the current evidence for each SHARE recommendation was presented to the expert committee. These recommendations were then discussed and revised during a video consensus meeting on January 22, 2021, with 14 voting participants, using a nominal group technique to reach consensus., Results: JIAU treatment was extended to include CAU. Fourteen recommendations regarding treatment of JIAU und CAU with >90% agreement were accepted., Conclusion: An update to the previous 2018 SHARE recommendations for the treatment of children with JIAU with the addition of CAU was created using an evidence-based consensus process. This guideline should help support clinicians to care for children and young people with CAU., (© 2022 American College of Rheumatology.)
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- 2023
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20. Discrete Choice Experiment on a Magnetic Resonance Imaging Scoring System for Temporomandibular Joints in Juvenile Idiopathic Arthritis.
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Tolend M, Junhasavasdikul T, Cron RQ, Inarejos Clemente EJ, von Kalle T, Kellenberger CJ, Koos B, Miller E, van Rossum MA, Saurenmann RK, Spiegel L, Stimec J, Twilt M, Tzaribachev N, Abramowicz S, Appenzeller S, Arvidsson LZ, Guleria S, Jaremko JL, Kirkhus E, Larheim TA, Meyers AB, Panwar J, Resnick CM, Shelmerdine SC, Feldman BM, and Doria AS
- Subjects
- Humans, Arthritis, Juvenile diagnostic imaging, Magnetic Resonance Imaging, Severity of Illness Index, Temporomandibular Joint diagnostic imaging
- Abstract
Objective: To determine the relative importance weights of items and grades of a newly developed additive outcome measure called the juvenile idiopathic arthritis (JIA) magnetic resonance imaging (MRI) scoring system for the temporomandibular joint (TMJ) (JAMRIS-TMJ)., Methods: An adaptive partial-profile, discrete choice experiment (DCE) survey using the 1000Minds platform was independently completed by members of an expert group consisting of radiologists and non-radiologist clinicians to determine the group-averaged relative weights for the JAMRIS-TMJ. Subsequently, an image-based vignette ranking exercise was done, during which experts individually rank ordered 14 patient vignettes for disease severity while blinded to the weights and unrestricted to JAMRIS-TMJ assessment criteria. Validity of the weighted JAMRIS-TMJ was tested by comparing the consensus-graded, DCE-weighted JAMRIS-TMJ score of the vignettes with their unrestricted image-based ranks provided by the experts., Results: Nineteen experts completed the DCE survey, and 21 completed the vignette ranking exercise. Synovial thickening and joint enhancement showed higher weights per raw score compared to bone marrow items and effusion in the inflammatory domain, while erosions and condylar flattening showed nonlinear and higher weights compared to disk abnormalities in the damage domain. The weighted JAMRIS-TMJ score of the vignettes correlated highly with the ranks from the unrestricted comparison method, with median Spearman's ρ of 0.92 (interquartile range [IQR] 0.87-0.95) for the inflammation and 0.93 (IQR 0.90-0.94) for the damage domain., Conclusion: A DCE survey was used to quantify the importance weights of the items and grades of the JAMRIS-TMJ. The weighted score showed high convergent validity with an unrestricted, holistic vignette ranking method., (© 2021, American College of Rheumatology.)
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- 2022
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21. Identification of an Amino Acid Motif in HLA-DRβ1 That Distinguishes Uveitis in Patients With Juvenile Idiopathic Arthritis.
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Haasnoot AJW, Schilham MW, Kamphuis S, Hissink Muller PCE, Heiligenhaus A, Foell D, Minden K, Ophoff RA, Radstake TRDJ, Den Hollander AI, Reinards THCM, Hiddingh S, Schalij-Delfos NE, Hoppenreijs EPAH, van Rossum MAJ, Wouters C, Saurenmann RK, van den Berg JM, Wulffraat NM, Ten Cate R, de Boer JH, Pulit SL, and Kuiper JJW
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- Adolescent, Alleles, Child, Child, Preschool, Cohort Studies, Female, Genetic Association Studies, Genetic Loci, Genotype, Humans, Male, Odds Ratio, Amino Acid Motifs genetics, Arthritis, Juvenile genetics, Genetic Predisposition to Disease genetics, HLA-DRB1 Chains genetics, Uveitis genetics
- Abstract
Objective: Uveitis is a visually debilitating disorder that affects up to 30% of children with the most common forms of juvenile idiopathic arthritis (JIA). The disease mechanisms predisposing only a subgroup of children to uveitis are unknown. This study was undertaken to identify genetic susceptibility loci for uveitis in JIA, using a genome-wide association study in 522 children with JIA., Methods: Two cohorts of JIA patients with ophthalmologic follow-up data were genotyped. Data were then imputed using a genome-wide imputation reference panel, and an HLA-specific reference panel was used for imputing amino acids and HLA types in the major histocompatibility complex (MHC). After imputation, genome-wide and MHC-specific analyses were performed, and a reverse immunology approach was utilized to model antigen presentation at 13 common HLA-DRβ1 alleles., Results: Presence of the amino acid serine at position 11 (serine 11) in HLA-DRβ1 was associated with an increased risk of uveitis in JIA patients (odds ratio [OR] 2.60, P = 5.43 × 10
-10 ) and was specific to girls (Pfemales = 7.61 × 10-10 versus Pmales = 0.18). Serine 11 resides in the YST motif in the peptide-binding groove of HLA-DRβ1; all 3 amino acids in this motif are in perfect linkage disequilibrium and show identical association with disease. Quantitative prediction of binding affinity revealed that HLA-DRβ1 alleles with the YST motif could be distinguished on the basis of discernable peptide-binding preferences., Conclusion: These findings highlight a genetically distinct, sexually dimorphic feature of JIA with uveitis as compared to JIA without uveitis. The association could be indicative of the potential involvement of antigen presentation by HLA-DRβ1 in the development of uveitis in JIA. The results of this study may advance our progress toward improved treatments for, and possible prevention of, the sight-threatening complications of uveitis in children with JIA., (© 2018, American College of Rheumatology.)- Published
- 2018
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22. Temporomandibular Joint Involvement in Association With Quality of Life, Disability, and High Disease Activity in Juvenile Idiopathic Arthritis.
- Author
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Frid P, Nordal E, Bovis F, Giancane G, Larheim TA, Rygg M, Pires Marafon D, De Angelis D, Palmisani E, Murray KJ, Oliveira S, Simonini G, Corona F, Davidson J, Foster H, Steenks MH, Flato B, Zulian F, Baildam E, Saurenmann RK, Lahdenne P, Ravelli A, Martini A, Pistorio A, and Ruperto N
- Subjects
- Adolescent, Arthritis, Juvenile physiopathology, Arthritis, Juvenile psychology, Case-Control Studies, Child, Child, Preschool, Cross-Sectional Studies, Female, Humans, Infant, Logistic Models, Male, Odds Ratio, Randomized Controlled Trials as Topic, Temporomandibular Joint physiopathology, Temporomandibular Joint Disorders etiology, Temporomandibular Joint Disorders physiopathology, Arthritis, Juvenile complications, Disability Evaluation, Quality of Life, Severity of Illness Index, Temporomandibular Joint Disorders psychology
- Abstract
Objective: To evaluate the demographic, disease activity, disability, and health-related quality of life (HRQOL) differences between children with juvenile idiopathic arthritis (JIA) and their healthy peers, and between children with JIA with and without clinical temporomandibular joint (TMJ) involvement and its determinants., Methods: This study is based on a cross-sectional cohort of 3,343 children with JIA and 3,409 healthy peers, enrolled in the Pediatric Rheumatology International Trials Organisation HRQOL study or in the methotrexate trial. Potential determinants of TMJ involvement included demographic, disease activity, disability, and HRQOL measures selected through univariate and multivariable logistic regression., Results: Clinical TMJ involvement was observed in 387 of 3,343 children with JIA (11.6%). Children with TMJ involvement, compared to those without, more often had polyarticular disease course (95% versus 70%), higher Juvenile Arthritis Disease Activity Score (odds ratio [OR] 4.6), more disability, and lower HRQOL. Children with TMJ involvement experienced clearly more disability and lower HRQOL compared to their healthy peers. The multivariable analysis showed that cervical spine involvement (OR 4.6), disease duration >4.4 years (OR 2.8), and having more disability (Childhood Health Assessment Questionnaire Disability Index >0.625) (OR 1.6) were the most important determinants for TMJ involvement., Conclusion: Clinical TMJ involvement in JIA is associated with higher disease activity, higher disability, and impaired HRQOL. Our findings indicate the need for dedicated clinical and imaging evaluation of TMJ arthritis, especially in children with cervical spine involvement, polyarticular course, and longer disease duration., (© 2016, American College of Rheumatology.)
- Published
- 2017
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23. Is early TMJ involvement in children with juvenile idiopathic arthritis clinically detectable? Clinical examination of the TMJ in comparison with contrast enhanced MRI in patients with juvenile idiopathic arthritis.
- Author
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Keller H, Müller LM, Markic G, Schraner T, Kellenberger CJ, and Saurenmann RK
- Subjects
- Adolescent, Arthritis, Juvenile pathology, Child, Child, Preschool, Female, Humans, Infant, Magnetic Resonance Imaging methods, Male, Physical Examination, Temporomandibular Joint pathology, Temporomandibular Joint Disorders etiology, Temporomandibular Joint Disorders pathology, Arthritis, Juvenile complications, Temporomandibular Joint Disorders diagnosis
- Abstract
Background: To test clinical findings associated with early temporomandibular joint (TMJ) arthritis in comparison to the current gold standard contrast enhanced magnetic resonance imaging (MRI) in children with juvenile idiopathic arthritis (JIA)., Methods: Seventy-six consecutive JIA patients were included in this study. Rheumatological and orthodontic examinations were performed blinded to MRI findings. Joint effusion and/or increased contrast enhancement of synovium or bone as well as TMJ deformity were assessed on MRI and compared to clinical findings. The maximal mouth opening capacity (MOC) of the JIA patients was compared to normative values obtained from a cohort of 20719 school children from Zürich, Switzerland., Results: On MRI a total of 54/76 (71%) patients and 92/152 (61%) joints had signs of TMJ involvement. MRI showed enhancement in 85/152 (56%) and deformity in 39/152 (26%) joints. MOC, asymmetry and restriction in condylar translation showed significant correlation to TMJ enhancement and deformity, whereas antegonial notching was correlated with TMJ deformity only. When joints with deformity were excluded, enhancement alone did not show a significant correlation with any clinical factor., Conclusions: Clinical findings in affected TMJs are correlated with structural damage only. Therefore clinical assessment of TMJs does not allow to diagnose early arthritis accurately and will still depend on contrast enhanced MRI.
- Published
- 2015
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24. Maximal mouth opening capacity: percentiles for healthy children 4-17 years of age.
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Müller L, van Waes H, Langerweger C, Molinari L, and Saurenmann RK
- Abstract
Background: A reduced mouth opening capacity may be one of the first clinical signs of pathological changes in the masticatory system. The aim of this retrospective cross-sectional study was to create age related percentiles for unassisted maximal mouth opening capacity (MOC) of healthy children., Methods: All recordings of MOC as measured at the yearly dental examinations of school children in the city of Zurich, Switzerland, between August 2009 and August 2010 were extracted from the database. The program LMSchartMaker Pro Version 2.43, Huiqi Pan and Tim Cole, Medical Research Council, 1997-2010 was used to calculate age and sex related reference centiles., Results: Records from 22(')060 dental examinations were found during the study period. In 1286 (5.8%) the maximal interincisal measurement was missing. Another 55 examinations were excluded because of missing data for sex (7), age at examination (11) or because the value was deemed to be pathologically low (37). Thus, a total of 20(')719 measurements (10(')060 girls, 10(')659 boys) were included in the analysis. The median age (range) was 9.9 years (3.3-18.3) for girls and 10.0 years (2.8-18.7) for boys. The mean MOC (range) was 45 mm (25-69) for girls and 45 mm (25-70) for boys. Age related percentiles were created for girls and boys separately, showing the 3(rd), 10(th), 25(th), 50(th), 75(th), 90(th), and 97(th) percentile from 3 through 18 years of age., Conclusions: In these 20(')719 unselected school children MOC increased with age but showed a wide range within children of the same age.
- Published
- 2013
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25. Juvenile Psoriatic Arthritis (JPsA): juvenile arthritis with psoriasis?
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Butbul Aviel Y, Tyrrell P, Schneider R, Dhillon S, Feldman BM, Laxer R, Saurenmann RK, Spiegel L, Cameron B, Tse SM, and Silverman E
- Abstract
Background: Following the introduction of the ILAR criteria for juvenile idiopathic arthritis, juvenile psoriatic arthritis (JPsA) has become a better recognized category within the inflammatory arthritides of childhood. There are fewer reports describing the characteristics and long-term outcome of patients with JPsA than other subtypes of JIA.The aim of our study was to determine the long-term outcome and clinical course of patients with juvenile psoriatic arthritis (JPsA) and to define subgroups of JPsA., Methods: Clinical records of all patients meeting criteria for JPsA were reviewed and divided into 4 groups depending on their clinical features and onset type. Patient characteristics and clinical features at onset and during follow-up were determined., Results: The cohort consisted of 119 patients: 65 with oligoarticular-onset (55%; persistent 44 and extended 21), 34 (29%) with RF(-) and 4 (3%) RF(+) polyarticular and 16 (13%) enthesitis-related arthritis (ERA). At diagnosis patients with ERA were oldest and more commonly male (p=0.001 and =0.01 respectively). Patients with a polyarticular course had more involvement of small joints of the hands and wrist when compared to patients with persistent oligoarticular and ERA (p<0.001) while patients with ERA had more hip and sacroiliac arthritis (p<0.001 for both). Nail changes were seen in 66 patients (57%) and were associated with DIP involvement (p=0.0034)., Outcome: Time to first inactive disease on, but not off, therapy was significantly longer among patients with polyarticular course when compared to oligoarticular and ERA (p=0.016 and p=0.48 respectively). Patients with polyarticular course more frequently had contractures during follow-up than other groups (p=0.01)., Conclusion: The long-term outcome of with JPsA was generally good. Patients with JPsA did not appear to form distinct sub-group of patients but rather resembled JIA patients with onset types without psoriasis.
- Published
- 2013
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26. Proposed outcome measures for prospective clinical trials in juvenile idiopathic arthritis-associated uveitis: a consensus effort from the multinational interdisciplinary working group for uveitis in childhood.
- Author
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Heiligenhaus A, Foeldvari I, Edelsten C, Smith JR, Saurenmann RK, Bodaghi B, de Boer J, Graham E, Anton J, Kotaniemi K, Mackensen F, Minden K, Nielsen S, Rabinovich EC, Ramanan AV, and Strand V
- Subjects
- Age Factors, Consensus, Cooperative Behavior, Delphi Technique, Endpoint Determination, Humans, Interdisciplinary Communication, International Cooperation, Longitudinal Studies, Predictive Value of Tests, Prospective Studies, Reproducibility of Results, Severity of Illness Index, Treatment Outcome, Uveitis diagnosis, Uveitis etiology, Arthritis, Juvenile complications, Randomized Controlled Trials as Topic standards, Uveitis therapy
- Abstract
Objective: To develop a set of core outcome measures for use in randomized controlled trials (RCTs) and longitudinal observational studies in juvenile idiopathic arthritis (JIA)-associated uveitis., Methods: The literature relating to outcome measures used in studies of uveitis in childhood and adolescence was reviewed. A set of core outcomes and domains was established using the Delphi process. This was reviewed by a representative multinational interdisciplinary working group. Nominal group technique consensus was reached on face and content validity of the range and content of the domains. The outcomes and the appropriate instruments for uveitis trials were adapted to the age ranges of patients with JIA-associated uveitis., Results: Consensus was reached that data should be reported at defined time points in longitudinal studies with patients stratified by prognostic markers. Visual acuity testing should be age appropriate. The severity of uveitis (measured as anterior chamber cell grade) and duration of active inflammation should be documented. Visually significant structural complications should be recorded and quantified with standard measures. The responses to treatment and corticosteroid-sparing effects of treatment should be documented. Patient-reported disease activity and age-specific uveitis-related quality of life should be reported using appropriate questionnaires., Conclusion: The proposed outcome measures in JIA-associated uveitis should aid in the standardization and comparison of future RCTs of the treatment regimens for this disease. The proposed outcome measures will be verified in a prospective validation study., (Copyright © 2012 by the American College of Rheumatology.)
- Published
- 2012
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27. Risk factors for development of uveitis differ between girls and boys with juvenile idiopathic arthritis.
- Author
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Saurenmann RK, Levin AV, Feldman BM, Laxer RM, Schneider R, and Silverman ED
- Subjects
- Adolescent, Age Factors, Age of Onset, Arthritis, Juvenile immunology, Chi-Square Distribution, Child, Child, Preschool, Databases, Factual, Female, Humans, Infant, Logistic Models, Male, Retrospective Studies, Risk Factors, Sex Factors, Uveitis immunology, Antibodies, Antinuclear immunology, Arthritis, Juvenile complications, Uveitis etiology
- Abstract
Objective: Uveitis is the most common extraarticular manifestation of juvenile idiopathic arthritis (JIA) and is associated with considerable morbidity. The aim of this study was to examine the risk factors associated with uveitis in JIA., Methods: We conducted a chart review of 1,047 patients with JIA from a single tertiary care pediatric rheumatology center for factors associated with the development of uveitis. Special emphasis was put on the following known risk factors: oligoarthritis, antinuclear antibody (ANA) status, sex, and age at the time of onset of JIA., Results: The risk of uveitis developing was age dependent in girls but not in boys. Among girls, the risk was maximal (47%) in those who were ANA positive and were ages 1-2 years at the time of the onset of JIA; this risk decreased to <10% in those in whom the age at onset was >7 years. Only girls had an age-dependent and ANA-associated increased risk of uveitis. The time interval from the diagnosis of JIA to the diagnosis of uveitis was statistically significantly longer in patients in whom the onset of JIA occurred at a younger age (P = 0.04). This effect was even more pronounced in ANA-positive patients (P = 0.004). The JIA subtype did not influence a patient's risk of the development of uveitis., Conclusion: An age-associated risk of uveitis was observed only in girls who were younger than 7 years of age at the time of the onset of JIA. The duration of time between the diagnosis of JIA and the onset of uveitis was longer in patients in whom JIA was diagnosed at a younger age, especially in those who were ANA positive. We suggest that our findings have implications for uveitis screening in patients with JIA.
- Published
- 2010
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28. Clinical course and prognostic value of disease activity in the first two years in different subtypes of juvenile idiopathic arthritis.
- Author
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Albers HM, Brinkman DM, Kamphuis SS, van Suijlekom-Smit LW, van Rossum MA, Hoppenreijs EP, Girschick HJ, Wouters C, Saurenmann RK, Houwing-Duistermaat JJ, Huizinga TW, Schilham MW, and ten Cate R
- Subjects
- Adolescent, Antirheumatic Agents therapeutic use, Arthritis physiopathology, Arthritis, Juvenile drug therapy, Child, Child, Preschool, Cohort Studies, Female, Humans, Infant, Male, Predictive Value of Tests, Prognosis, Remission Induction, Retrospective Studies, Time Factors, Arthritis, Juvenile classification, Arthritis, Juvenile physiopathology
- Abstract
Objective: Juvenile idiopathic arthritis (JIA) is a heterogeneous disease involving chronic arthritis. The clinical course is characterized by a fluctuating pattern of active and inactive disease. We have described in detail the clinical course in different JIA subtypes during the first 2 years after diagnosis and studied its relationship to disease activity in the following years., Methods: Detailed clinical data on different parameters describing the disease activity in sequential time periods covering the first 2 years after diagnosis were retrieved from the charts of 311 patients with JIA and compared between subtypes. In a cohort of 146 patients, the relation of these different clinical variables to the course of disease in the following 3 years was evaluated., Results: The percentage of time with active disease in the first 2 years differed significantly between subtypes. In all subtypes, a broad spectrum of activity was observed. The time with active disease in the first 2 years was the most significant factor associated with the duration of active disease in the following years., Conclusion: Different percentages of time with active disease have been observed between JIA subtypes in the first 2 years. The cumulative duration of activity varied widely within each subtype. Regarding the prognosis of the individual patient, the clinical course in the first 2 years appears to be predictive of the clinical course in the following years. Patients that have less time with active disease in the first 2 years are not likely to develop an unremitting clinical course later on.
- Published
- 2010
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29. Current educational status of paediatric rheumatology in Europe: the results of PReS survey.
- Author
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Demirkaya E, Ozen S, Türker T, Kuis W, and Saurenmann RK
- Subjects
- Adolescent, Child, Europe, Health Policy, Humans, Interprofessional Relations, Pediatrics trends, Rheumatology trends, Societies, Medical, Surveys and Questionnaires, Education, Medical, Graduate, Pediatrics education, Rheumatology education
- Abstract
Objectives: To understand the status of education and problems in paediatric rheumatology practice in Europe, through a survey., Methods: A 26-item questionnaire was conducted during the 14th Congress of the Paediatric Rheumatology European Society in Istanbul, 2007. Physicians who were practicing or studying within the field of paediatric rheumatology for at least one year were included in the survey., Results: One hundred and twenty eight physicians, 79 paediatric rheumatologists (including 5 paediatric immunologists and 10 paediatric nephrologists), 34 paediatric rheumatology fellows and 15 adult rheumatologists completed the survey. The physicians were from: Europe 95 (81.9%), South America 12 (10.4%), Middle East 5 (4.3%), Asia 2 (1.7%), Africa 2 (1.7%). The duration of training for paediatric rheumatology ranged between 1-5 years (mean: 3.12+/-1.11). Sixty physicians scored their education as unsatisfactory and among those, 48 physicians were from Europe. Physicians reported good skills in the following items; intraarticular injections (83.3%); soft tissue injections (47.6%); evaluation of radiographs (67.5%); whereas competence in the evaluation of computed tomography/magnetic resonance imaging (30.5%); and musculoskeletal sonography (16.7%) was much lower. A need for improved basic science and rotations among relevant fields were specifically expressed., Conclusion: Being a relatively new speciality in the realm of paediatrics, paediatric rheumatology education at the European level needs to be further discussed, revised and uniformed.
- Published
- 2009
30. Early diagnosis of temporomandibular joint involvement in juvenile idiopathic arthritis: a pilot study comparing clinical examination and ultrasound to magnetic resonance imaging.
- Author
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Müller L, Kellenberger CJ, Cannizzaro E, Ettlin D, Schraner T, Bolt IB, Peltomäki T, and Saurenmann RK
- Subjects
- Adolescent, Arthritis, Juvenile diagnostic imaging, Chi-Square Distribution, Child, Child, Preschool, Early Diagnosis, Female, Humans, Infant, Male, Orthodontics, Physical Examination, Pilot Projects, Rheumatology, Sensitivity and Specificity, Temporomandibular Joint diagnostic imaging, Temporomandibular Joint Disorders diagnostic imaging, Ultrasonography, Arthritis, Juvenile diagnosis, Magnetic Resonance Imaging, Temporomandibular Joint pathology, Temporomandibular Joint Disorders diagnosis
- Abstract
Objectives: To study the validity of both rheumatological and orthodontic examinations and ultrasound (US) as screening methods for early diagnosis of TMJ arthritis against the gold standard MRI., Methods: Thirty consecutive juvenile idiopathic arthritis (JIA) patients were included in this pilot study. Rheumatological and orthodontic examinations as well as US were performed within 1 month of the MRI in a blinded fashion. Joint effusion and/or increased contrast enhancement of synovium or bone were considered signs of active arthritis on MRI., Results: A total of 19/30 (63%) patients and 33/60 (55%) joints had signs of TMJ involvement on MRI. This was associated with condylar deformity in 9/19 (47%) patients and 15/33 (45%) joints. Rheumatological, orthodontic and US examinations correctly diagnosed 11 (58%), 9 (47%) and 6 (33%) patients, respectively, with active TMJ arthritis, but misdiagnosed 8 (42%), 10 (53%) and 12 (67%) patients, respectively, as having no signs of inflammation. The best predictor for active arthritis on MRI was a reduced maximum mouth opening., Conclusion: None of the methods tested was able to reliably predict the presence or absence of MRI-proven inflammation in the TMJ in our cohort of JIA patients. US was the least useful of all methods tested to exclude active TMJ arthritis.
- Published
- 2009
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31. The provisional Paediatric Rheumatology International Trials Organisation/American College of Rheumatology/European League Against Rheumatism Disease activity core set for the evaluation of response to therapy in juvenile dermatomyositis: a prospective validation study.
- Author
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Ruperto N, Ravelli A, Pistorio A, Ferriani V, Calvo I, Ganser G, Brunner J, Dannecker G, Silva CA, Stanevicha V, Cate RT, van Suijlekom-Smit LW, Voygioyka O, Fischbach M, Foeldvari I, Hilario O, Modesto C, Saurenmann RK, Sauvain MJ, Scheibel I, Sommelet D, Tambic-Bukovac L, Barcellona R, Brik R, Ehl S, Jovanovic M, Rovensky J, Bagnasco F, Lovell DJ, and Martini A
- Subjects
- Child, Female, Humans, Male, Prospective Studies, Dermatomyositis diagnosis, Practice Guidelines as Topic
- Abstract
Objective: To validate a core set of outcome measures for the evaluation of response to treatment in patients with juvenile dermatomyositis (DM)., Methods: In 2001, a preliminary consensus-derived core set for evaluating response to therapy in juvenile DM was established. In the present study, the core set was validated through an evidence-based, large-scale data collection that led to the enrollment of 294 patients from 36 countries. Consecutive patients with active disease were assessed at baseline and after 6 months. The validation procedures included assessment of feasibility, responsiveness, discriminant and construct ability, concordance in the evaluation of response to therapy between physicians and parents, redundancy, internal consistency, and ability to predict a therapeutic response., Results: The following clinical measures were found to be feasible, and to have good construct validity, discriminative ability, and internal consistency; furthermore, they were not redundant, proved responsive to clinically important changes in disease activity, and were associated strongly with treatment outcome and thus were included in the final core set: 1) physician's global assessment of disease activity, 2) muscle strength, 3) global disease activity measure, 4) parent's global assessment of patient's well-being, 5) functional ability, and 6) health-related quality of life., Conclusion: The members of the Paediatric Rheumatology International Trials Organisation, with the endorsement of the American College of Rheumatology and the European League Against Rheumatism, propose a core set of criteria for the evaluation of response to therapy that is scientifically and clinically relevant and statistically validated. The core set will help standardize the conduct and reporting of clinical trials and assist practitioners in deciding whether a child with juvenile DM has responded adequately to therapy.
- Published
- 2008
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32. A randomized, placebo-controlled trial of infliximab plus methotrexate for the treatment of polyarticular-course juvenile rheumatoid arthritis.
- Author
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Ruperto N, Lovell DJ, Cuttica R, Wilkinson N, Woo P, Espada G, Wouters C, Silverman ED, Balogh Z, Henrickson M, Apaz MT, Baildam E, Fasth A, Gerloni V, Lahdenne P, Prieur AM, Ravelli A, Saurenmann RK, Gamir ML, Wulffraat N, Marodi L, Petty RE, Joos R, Zulian F, McCurdy D, Myones BL, Nagy K, Reuman P, Szer I, Travers S, Beutler A, Keenan G, Clark J, Visvanathan S, Fasanmade A, Raychaudhuri A, Mendelsohn A, Martini A, and Giannini EH
- Subjects
- Antibodies, Monoclonal adverse effects, Antirheumatic Agents adverse effects, Child, Double-Blind Method, Drug Therapy, Combination, Female, Humans, Infliximab, Male, Methotrexate adverse effects, Antibodies, Monoclonal administration & dosage, Antirheumatic Agents administration & dosage, Arthritis, Juvenile drug therapy, Methotrexate administration & dosage
- Abstract
Objective: To evaluate the safety and efficacy of infliximab in the treatment of juvenile rheumatoid arthritis (JRA)., Methods: This was an international, multicenter, randomized, placebo-controlled, double-blind study. One hundred twenty-two children with persistent polyarticular JRA despite prior methotrexate (MTX) therapy were randomized to receive infliximab or placebo for 14 weeks, after which all children received infliximab through week 44. Patients received MTX plus infliximab 3 mg/kg through week 44, or MTX plus placebo for 14 weeks followed by MTX plus infliximab 6 mg/kg through week 44., Results: Although a higher proportion of patients in the 3 mg/kg infliximab group than in the placebo group had achieved responses according to the American College of Rheumatology (ACR) Pediatric 30 (Pedi 30) criteria for improvement at week 14 (63.8% and 49.2%, respectively), the between-group difference in this primary efficacy end point was not statistically significant (P = 0.12). By week 16, after the crossover from placebo to infliximab 6 mg/kg when all patients were receiving infliximab, an ACR Pedi 30 response was achieved in 73.2% of all patients. By week 52, ACR Pedi 50 and ACR Pedi 70 responses had been reached in 69.6% and 51.8%, respectively, of patients. Infliximab was generally well tolerated, but the safety profile of infliximab 3 mg/kg appeared less favorable than that of infliximab 6 mg/kg, with more frequent occurrences of serious adverse events, infusion reactions, antibodies to infliximab, and newly induced antinuclear antibodies and antibodies to double-stranded DNA observed with the 3 mg/kg dose., Conclusion: While infliximab at 3 mg/kg and 6 mg/kg showed durable efficacy at 1 year, achievement of the primary efficacy end point at 3 months did not differ significantly between infliximab-treated and placebo-treated patients. Safety data indicated that the 6-mg/kg dose may provide a more favorable risk/benefit profile. These results warrant further investigation in children with JRA.
- Published
- 2007
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33. Tumour necrosis factor alpha inhibitors in the treatment of childhood uveitis.
- Author
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Saurenmann RK, Levin AV, Rose JB, Parker S, Rabinovitch T, Tyrrell PN, Feldman BM, Laxer RM, Schneider R, and Silverman ED
- Subjects
- Adolescent, Arthritis, Juvenile complications, Child, Child, Preschool, Drug Evaluation, Etanercept, Female, Humans, Infant, Infliximab, Male, Retrospective Studies, Treatment Outcome, Uveitis etiology, Antibodies, Monoclonal therapeutic use, Antirheumatic Agents therapeutic use, Immunoglobulin G therapeutic use, Receptors, Tumor Necrosis Factor therapeutic use, Tumor Necrosis Factor-alpha antagonists & inhibitors, Uveitis drug therapy
- Abstract
Objective: To describe the efficacy of anti-TNF-alpha agents in the treatment of childhood uveitis., Methods: We performed a retrospective chart review of all children with uveitis treated with TNF-alpha blockers at The Hospital for Sick Children, Toronto., Results: Twenty-one children with uveitis were treated with the anti-TNF-alpha agents etanercept (11 patients) and infliximab (13 patients), resulting in 24 treatment courses. All patients had persistently active uveitis despite treatment with at least one standard immunosuppressive drug before the start of anti-TNF-alpha therapy. Six of 21 patients (29%) had idiopathic uveitis. In the other 15 patients, the underlying disease was juvenile idiopathic arthritis in 12 (57%), Behçet disease in two (9%) and sarcoidosis in one (5%). Response to etanercept treatment was good in 27%, moderate in 27% and poor in 45% of patients. Response to infliximab treatment was good in 38%, moderate in 54% and poor in 8% of patients. The difference in the percentage of patients with a moderate or good response was statistically significant (P = 0.0481). We also observed a lower rate of complications, such as new-onset or worsening glaucoma or cataract in the infliximab-treated group., Conclusion: Anti-TNF-alpha treatment was beneficial in a high percentage of patients with childhood uveitis refractory to standard immunosuppressive treatment. Infliximab resulted in better clinical responses with less ocular complications than etanercept.
- Published
- 2006
- Full Text
- View/download PDF
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