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5. Defining anti-synthetase syndrome: a systematic literature review

Author

Zanframundo, G, Faghihi-Kashani, S, Scire, C, Bonella, F, Corte, T, Doyle, T, Fiorentino, D, Gonzalez-Gay, M, Hudson, M, Kuwana, M, Lundberg, I, Mammen, A, Mchugh, N, Miller, F, Monteccucco, C, Oddis, C, Rojas-Serrano, J, Schmidt, J, Selva-O'Callaghan, A, Werth, V, Sakellariou, G, Aggarwal, R, Cavagna, L, Zanframundo G., Faghihi-Kashani S., Scire C. A., Bonella F., Corte T. J., Doyle T. J., Fiorentino D., Gonzalez-Gay M. A., Hudson M., Kuwana M., Lundberg I. E., Mammen A., McHugh N., Miller F. W., Monteccucco C., Oddis C. V., Rojas-Serrano J., Schmidt J., Selva-O'Callaghan A., Werth V. P., Sakellariou G., Aggarwal R., Cavagna L., Zanframundo, G, Faghihi-Kashani, S, Scire, C, Bonella, F, Corte, T, Doyle, T, Fiorentino, D, Gonzalez-Gay, M, Hudson, M, Kuwana, M, Lundberg, I, Mammen, A, Mchugh, N, Miller, F, Monteccucco, C, Oddis, C, Rojas-Serrano, J, Schmidt, J, Selva-O'Callaghan, A, Werth, V, Sakellariou, G, Aggarwal, R, Cavagna, L, Zanframundo G., Faghihi-Kashani S., Scire C. A., Bonella F., Corte T. J., Doyle T. J., Fiorentino D., Gonzalez-Gay M. A., Hudson M., Kuwana M., Lundberg I. E., Mammen A., McHugh N., Miller F. W., Monteccucco C., Oddis C. V., Rojas-Serrano J., Schmidt J., Selva-O'Callaghan A., Werth V. P., Sakellariou G., Aggarwal R., and Cavagna L.

Abstract

Anti-synthetase syndrome (ASSD) is a heterogeneous autoimmune disease characterised by multi-system involvement with a wide variety of manifestations. Validated classification criteria are necessary to improve recognition and prevent misclassification, especially given the lack of reliable and standardised autoantibody testing. We systematically reviewed the literature to analyse proposed ASSD criteria, characteristics, and diagnostic performance. Methods We searched PubMed and Embase databases (01/01/1984 to 06/11/2018) and the ACR and EULAR meeting abstracts (2017-2018). Sensitivities, specificities, positive, negative likelihood ratios and risk of bias were calculated for ASSD criteria and key variables reported in the literature. We performed meta-analysis when appropriate. Results We retrieved 4,358 studies. We found 85 proposed ASSD criteria from a total of 82 studies. All but one study included anti-synthetase autoantibody (ARS) positivity in the ASSD criteria. Most studies required only one ASSD feature plus anti-ARS to define ASSD (n=64, 78%), whereas 16 studies required more than one ASSD variable plus anti-ARS. The only criteria not including anti-ARS positivity required 5 ASSD clinical features. We found limited data and wide variability in the diagnostic performance of each variable and definition proposed in the literature. Given these limitations we only meta-analysed the performance of individual muscle biopsy and clinical variables in diagnosing ASSD, which performed poorly. Conclusion The current ASSD criteria include a variety of serological, clinical, and histological features with wide variability amongst proposed definitions and the performance of these definitions has not been tested. This systematic literature review suggests the need for additional data and consensus-driven classification criteria for ASSD.

Published
2022

7. POS0143 TISSUE RESIDENT MEMORY AND CYTOTOXIC T CELLS ARE CLONALLY EXPANDED IN THE MUSCLE OF PATIENTS WITH IDIOPATHIC INFLAMMATORY MYOPATHIES

Author

Galindo-Feria, A. S., primary, Argyriou, A., additional, Horuluoglu, B., additional, Díaz-Boada, J. S., additional, Notarnicola, A., additional, Dani, L., additional, Van Vollenhoven, A., additional, Ramsköld, D., additional, Nennesmo, I., additional, Dastmalchi, M., additional, Lundberg, I. E., additional, Diaz-Gallo, L. M., additional, and Chemin, K., additional

Published
2023
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11. POS0603 AUTOANTIBODIES AGAINST A SUBUNIT OF MITOCHONDRIAL RESPIRATORY CHAIN COMPLEX I IN INCLUSION BODY MYOSITIS

Author

Notarnicola, A., primary, Hellstrom, C., additional, Horuluoglu, B., additional, Preger, C., additional, Bonomi, F., additional, De Paepe, B., additional, De Bleecker, J., additional, Van der Kooi, A. J., additional, De Visser, M., additional, Sacconi, S., additional, Machado, P., additional, Badrising, U. A., additional, Rietveld, A., additional, Pruijn, G., additional, Rothwell, S., additional, Lilleker, J. B., additional, Chinoy, H., additional, Benveniste, O., additional, Svenungsson, E., additional, Idborg, H., additional, Jakobsson, P. J., additional, Nilsson, P., additional, and Lundberg, I. E., additional

Published
2023
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15. POS1221 CLINICAL FEATURES OF PATIENTS WITH ANTI-SYNTHETASE SYNDROME AND DERMATOMYOSITIS-ASSOCIATED SKIN MANIFESTATIONS: RESULTS FROM THE MYONET REGISTRY

Author

Hum, R. M., primary, Lilleker, J. B., additional, Lamb, J., additional, Oldroyd, A., additional, Ollier, W., additional, Wang, G., additional, Kodishala, C., additional, Wedderburn, L., additional, Diederichsen, L., additional, Schmidt, J., additional, Danieli, M. G., additional, Dankó, K., additional, Nguyen, T. P. T., additional, Vázquez-Del Mercado Espinosa, M., additional, Andersson, H., additional, De Paepe, B., additional, De Bleecker, J., additional, Maurer, B., additional, Mccann, L., additional, Pipitone, N., additional, New, R. P., additional, Steen Krogh, N., additional, Mchugh, N., additional, Vencovský, J., additional, Lundberg, I. E., additional, and Chinoy, H., additional

Published
2023
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16. Evaluation of an individually tailored smoking-cessation intervention for patients with rheumatoid arthritis in an outpatient clinic

Author

Karlsson, M-l, Hertzberg-Nyquist, K., Saevarsdottir, S., Lundberg, I. E., Demmelmaier, Ingrid, Pettersson, S., Chatzidionysiou, K., Karlsson, M-l, Hertzberg-Nyquist, K., Saevarsdottir, S., Lundberg, I. E., Demmelmaier, Ingrid, Pettersson, S., and Chatzidionysiou, K.

Abstract

Objective: The aim of this study was to evaluate an individually tailored smoking-cessation intervention delivered in rheumatology care and compare the characteristics of patients who quit smoking with those who did not. Method: This was an open single-group prospective intervention study over 24 months, with assessments at baseline and at 6, 12, 18, and 24 months. Current smokers with rheumatoid arthritis (RA) were invited to a smoking-cessation programme including behavioural change support, with or without pharmacotherapy. Data on disease activity, medical treatment, and patient-reported outcomes were retrieved from the Swedish Rheumatology Quality Register. The primary outcome was the proportion of patients at month 24 who reported having quit smoking with self-reported 7 day smoking abstinence. Results: In total, 99 patients participated in the study. Median age was 58 years (interquartile range 50-64); 69% were female and 88% rheumatoid factor and/or anti-cyclic citrullinated peptide positive. At 24 months, 21% of the patients had quit smoking. At 6, 12, and 18 months, 12%, 12%, and 14% of patients, respectively, had quit smoking. For patients still smoking at 24 months, the median number of cigarettes per day was significantly reduced from 12 to 6 (p <= 0.001). Among patients who had quit smoking at 24 months, a smaller proportion reported anxiety at baseline compared to those still smoking (28% vs 58%, p = 0.02). Conclusion: A smoking-cessation intervention including behavioural change support with or without pharmacotherapy can be helpful for a substantial number of RA patients. Anxiety is associated with lower smoking-cessation success rates.

Published
2023
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18. Genome-wide association study identifies HLA 8.1 ancestral haplotype alleles as major genetic risk factors for myositis phenotypes

Author

Miller, F W, Chen, W, O'Hanlon, T P, Cooper, R G, Vencovsky, J, Rider, L G, Danko, K, Wedderburn, L R, Lundberg, I E, Pachman, L M, Reed, A M, Ytterberg, S R, Padyukov, L, Selva-O'Callaghan, A, Radstake, T R, Isenberg, D A, Chinoy, H, Ollier, W E R, Scheet, P, Peng, B, Lee, A, Byun, J, Lamb, J A, Gregersen, P K, and Amos, C I

Published
2015
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21. OP0160 HLA-DRB1 ASSOCIATIONS WITH AUTOANTIBODY-DEFINED SUBGROUPS IN IDIOPATHIC INFLAMMATORY MYOPATHIES (IIM)

Author

Leclair, V., primary, Galindo-Feria, A. S., additional, Rothwell, S., additional, Kryštůfková, O., additional, Mann, H., additional, Pyndt Diederichsen, L., additional, Andersson, H., additional, Klein, M., additional, Tansley, S., additional, Mchugh, N., additional, Lamb, J., additional, Vencovský, J., additional, Chinoy, H., additional, Holmqvist, M., additional, Padyukov, L., additional, Lundberg, I. E., additional, and Diaz-Gallo, L. M., additional

Published
2022
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24. Identification of Novel Associations and Localization of Signals in Idiopathic Inflammatory Myopathies Using Genome-Wide Imputation

Author

Rothwell, S., Amos, C. I., Miller, F. W., Rider, L. G., Lundberg, I. E., Gregersen, P. K., Vencovsky, J., Mchugh, N., Limaye, V., Selva-O'Callaghan, A., Hanna, M. G., Machado, P. M., Pachman, L. M., Reed, A. M., Molberg, Ø., Benveniste, O., Mathiesen, P., Radstake, T., Doria, A., De Bleecker, J. L., De Paepe, B., Maurer, B., Ollier, W. E., Padyukov, L., O'Hanlon, T. P., Lee, A., Wedderburn, L. R., Chinoy, H., Lamb, J. A., Institut Català de la Salut, [Rothwell S] Centre for Genetics and Genomics Versus Arthritis, Centre for Musculoskeletal Research, Faculty of Biology, Medicine and Health, University of Manchester, Manchester, UK. [Amos CI] Baylor College of Medicine, Houston, Texas. [Miller FW, Rider LG] Environmental Autoimmunity Group, National Institute of Environmental Health Sciences, NIH, Bethesda, Maryland. [Lundberg IE] Division of Rheumatology, Department of Medicine, Solna, Karolinska Institutet, Karolinska University Hospital, Stockholm, Sweden. [Gregersen PK] The Robert S. Boas Center for Genomics and Human Genetics, The Feinstein Institute, Manhasset, New York. [Selva-O'Callaghan A] Servei de Medicina Interna, Vall d’Hebron Hospital Universitari, Barcelona, Spain. Universitat Autònoma de Barcelona, Bellaterra, Spain, and Vall d'Hebron Barcelona Hospital Campus

Subjects
Rheumatology, Músculs - Malalties - Aspectes genètics, Otros calificadores::Otros calificadores::/genética [Otros calificadores], Immunology, Malalties autoimmunitàries - Aspectes genètics, Immune System Diseases::Autoimmune Diseases [DISEASES], Other subheadings::Other subheadings::/genetics [Other subheadings], Genetic Phenomena::Genotype::Genetic Predisposition to Disease [PHENOMENA AND PROCESSES], enfermedades del sistema inmune::enfermedades autoinmunes [ENFERMEDADES], Immunology and Allergy, Musculoskeletal Diseases::Muscular Diseases::Myositis [DISEASES], enfermedades musculoesqueléticas::enfermedades musculares::miositis [ENFERMEDADES], fenómenos genéticos::genotipo::predisposición genética a la enfermedad [FENÓMENOS Y PROCESOS]
Abstract

Idiopathic inflammatory myopathies; Genome Miopatías inflamatorias idiopáticas; Genoma Miopaties inflamatòries idiopàtiques; Genoma Objective The idiopathic inflammatory myopathies (IIMs) are heterogeneous diseases thought to be initiated by immune activation in genetically predisposed individuals. We imputed variants from the ImmunoChip array using a large reference panel to fine-map associations and identify novel associations in IIM. Methods We analyzed 2,565 Caucasian IIM patient samples collected through the Myositis Genetics Consortium (MYOGEN) and 10,260 ethnically matched control samples. We imputed 1,648,116 variants from the ImmunoChip array using the Haplotype Reference Consortium panel and conducted association analysis on IIM and clinical and serologic subgroups. Results The HLA locus was consistently the most significantly associated region. Four non-HLA regions reached genome-wide significance, SDK2 and LINC00924 (both novel) and STAT4 in the whole IIM cohort, with evidence of independent variants in STAT4, and NAB1 in the polymyositis (PM) subgroup. We also found suggestive evidence of association with loci previously associated with other autoimmune rheumatic diseases (TEC and LTBR). We identified more significant associations than those previously reported in IIM for STAT4 and DGKQ in the total cohort, for NAB1 and FAM167A-BLK loci in PM, and for CCR5 in inclusion body myositis. We found enrichment of variants among DNase I hypersensitivity sites and histone marks associated with active transcription within blood cells. Conclusion We found novel and strong associations in IIM and PM and localized signals to single genes and immune cell types. Supported by the Intramural Research Program, National Institute of Environmental Health Sciences, NIH. Dr. Lundberg's work was supported by grants from the Swedish Research Council and Stockholm Regional Council (ALF). Dr. Vencovsky's work was supported by the Czech Ministry of Health–Conceptual Development of Research Organization (award 00023728) (Institute of Rheumatology). Drs. Hanna and Machado's work were supported by the NIHR University College London Hospitals Biomedical Research Centre. Drs. De Bleecker and De Paepe's work were supported by the European Reference Network for Rare Neuromuscular Diseases (ERN EURO-NMD). Dr. Wedderburn's work was supported by Versus Arthritis (awards 21593 and 21552), the Wellcome trust (award 085860), Myositis UK, the Cure JM Foundation, the Remission Charity, and the NIHR Biomedical research Centre at GOSH. Dr. Chinoy's work was supported by the Medical Research Council UK (award MR/N003322/1), Myositis UK, and the NIHR Manchester Biomedical Research Centre Funding Scheme. Dr. Lamb's work was supported by the Medical Research Council UK (award MR/N003322/1) and Myositis UK.

Published
2022

27. The Role of Exercise to Improve Physiological, Physical and Psychological Health Outcome in Idiopathic Inflammatory Myopathies (IIM)

Author

Lodin K, Espinosa-Ortega F, Lundberg IE, and Alexanderson H

Subjects
exercise, idiopathic inflammatory myopathies, treatment, Pathology, RB1-214, Therapeutics. Pharmacology, RM1-950
Abstract

Karin Lodin,1,2,&ast; Fabricio Espinosa-Ortega,1,2,&ast; Ingrid E Lundberg,1,2 Helene Alexanderson1,3 1Division of Rheumatology, Department of Medicine Solna, Karolinska Institutet, Stockholm, Sweden; 2Department of Gastro, Dermatology and Rheumatology, Theme Inflammation and Aging, Karolinska University Hospital, Stockholm, Sweden; 3Medical Unit Allied Health Professionals, Theme Women’s Health and Health Professionals, Karolinska University Hospital, Stockholm, Sweden&ast;These authors contributed equally to this workCorrespondence: Helene Alexanderson, Medical Unit Occupational Therapy and Physical Therapy, Theme Women’s Health and Health Professionals, Karolinska University Hospital, Gävlegatan 55, NB02:00, Stockholm, 17176, Sweden, Email helene.alexanderson@ki.seAbstract: Idiopathic inflammatory myopathies (IIM) impact all aspects of health, physiological, physical, and psychological. Hallmark symptoms of IIM are muscle weakness, reduced muscle endurance and aerobic capacity. Recently, pain and fatigue as well as anxiety and depression have emerged as common and debilitating symptoms in patients with IIM. The aim of this scoping review is to, in a holistic way, describe how IIM impact patients’ physiological, physical, and psychological health and how exercise has a role to treat as well as potentially counteract the effects of the disease. Inflammation induces non-immune response and organ damage. These changes with additional impact of physical inactivity lead to muscle impairment and reduced aerobic capacity. Pain, fatigue and low psychological well-being and overall quality of life are also common health aspects of IIM. Medical treatment can reduce inflammation but has in turn serious side effects such as muscle atrophy, type-II diabetes, and hypertension, which exercise has the potential to treat, and perhaps also counteract. In addition, exercise improves muscle function, aerobic capacity and might also reduce fatigue and pain. New evidence shows that reducing systemic inflammation may also improve patient-reported subjective health, quality of life and psychological well-being. Exercise in combination with medical treatment is becoming an important part of the treatment for patients with IIM as exercise has the potential to promote health aspects of various dimensions in patients with IIM.Keywords: exercise, idiopathic inflammatory myopathies, treatment

Published
2024

28. Myositis-specific and myositis-associated autoantibodies predict trajectories of damage over time in idiopathic inflammatory myopathies

Author

Espinosa-Ortega, F., Holmqvist, M., Lodin, K., Dastmalchi, M., Danieli, M. G., Venkovsky, J., Knitza, J., Oliveira, S., Diederichsen, L. P., Wedderburn, L., Andersson, H., del Mercado, M. Vazquez, Gomez-Martin, D., Torres, J., Callaghan, A. Selva O., Benveniste, O., De Visser, M., De Bleecker, J., McCann, L., De Langhe, E., Rietveld, A., Chinoy, H., Krogh, N. S., Alexanderson, H., Lundberg, I. E., and Grp, Euro-myositis

Published
2021
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31. Autoantigenic properties indicated for the entire aminoacyl tRNA synthetase family in idiopathic inflammatory myopathies

Author

Preger, C., Notarnicola, A., Hellström, Cecilia, Wigren, E., Fernandes-Cerqueira, C., Nilsson, Peter, Lundberg, I. E., Persson, Helena, Graslund, S., Jakobsson, P. J., Preger, C., Notarnicola, A., Hellström, Cecilia, Wigren, E., Fernandes-Cerqueira, C., Nilsson, Peter, Lundberg, I. E., Persson, Helena, Graslund, S., and Jakobsson, P. J.

Abstract

QC 20210928

Published
2021

36. Serum and balf-derived anti-JO1 autoantibodies exhibit high reactivity to distinct HISRS domains and associate with lung and joint involvement in patients with IIM/ASS

Author

Notarnicola, A., Preger, C., Lundström, S., Renard, N., Wigren, E., Van Gompel, E., Galindo-Feria, A. S., Persson, Helena, Fathi, M., Grunewald, J., Jakobsson, P. J., Gräslund, S., Lundberg, I. E., Cerqueira, C., Notarnicola, A., Preger, C., Lundström, S., Renard, N., Wigren, E., Van Gompel, E., Galindo-Feria, A. S., Persson, Helena, Fathi, M., Grunewald, J., Jakobsson, P. J., Gräslund, S., Lundberg, I. E., and Cerqueira, C.

Abstract

QC 20200825

Published
2020
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37. Characterization of anti-aminoacyl TRNA synthetase autoantibodies in patients with idiopathic inflammatory myopathies

Author

Preger, C., Notarnicola, A., Hellström, Cecilia, Wigren, E., Cerqueira, C., Nilsson, Peter, Lundberg, I. E., Persson, Helena, Graslund, S., Jakobsson, P. J., Preger, C., Notarnicola, A., Hellström, Cecilia, Wigren, E., Cerqueira, C., Nilsson, Peter, Lundberg, I. E., Persson, Helena, Graslund, S., and Jakobsson, P. J.

Abstract

QC 20200903

Published
2020
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38. SAT0335 SERUM AND BALF-DERIVED ANTI-JO1 AUTOANTIBODIES EXHIBIT HIGH REACTIVITY TO DISTINCT HISRS DOMAINS AND ASSOCIATE WITH LUNG AND JOINT INVOLVEMENT IN PATIENTS WITH IIM/ASS

Author

Notarnicola, A., primary, Preger, C., additional, Lundström, S., additional, Renard, N., additional, Wigren, E., additional, Van Gompel, E., additional, Galindo-Feria, A. S., additional, Persson, H., additional, Fathi, M., additional, Grunewald, J., additional, Jakobsson, P. J., additional, Gräslund, S., additional, Lundberg, I. E., additional, and Cerqueira, C., additional

Published
2020
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42. 2016 ACR-EULAR adult dermatomyositis and polymyositis and juvenile dermatomyositis response criteria—methodological aspects

Author

Rider, L. G., Ruperto, N., Pistorio, A., Erman, B., Bayat, N., Lachenbruc, P. A., Rockette, H., Feldman, B. M., Huber, A. M., Hansen, P., Oddis, C. V., Lundberg, I. E., Amato, A. A., Chinoy, H., Cooper, R. G., Chung, L., Danko, K., Fiorentino, D., De la Torre, I. G., Reed, A. M., Song, Y. W., Cimaz, R., Cuttica, R. J., Pilkington, C. A., Martini, A., van der Net, J., Maillard, S., Miller, F. W., Vencovsky, J., Aggarwal, R., Christopher-Stine, L., Criscione-Schreiber, L., Crofford, L., Cronin, M. E., Gordon, P., Hengstman, G., Katz, J. D., Mammen, A., Marder, G., Mchugh, N., Schiopu, E., Wolfe, G., Wortmann, R., Apaz, M., Bowyer, S., Constantin, T., Curran, M., Davidson, J., Griffin, T., Jones, O., Kim, S., Lang, B., Lindsley, C., Lovell, D., Magalhaes, C. S., Pachman, L. M., Ponyi, A., Punaro, M., Quartier, P., Ramanan, A. V., Ravelli, A., Rennebohm, R., Van Royen-Kerkhof, A., Sherry, D. D., Silva, C. A., Stringer, E., Wallace, C., Ascherman, D., Barohn, R., Benveniste, O., De Bleecker, J., Callen, J., Charles-Schoeman, C., Danoff, S., Dastmalchi, M., Dimachkie, M., Di Martino, S., Dourmishev, L., Ernste, F., Gono, T., Isenberg, D., Katsumata, Y., Kissel, J., Leff, R. L., Levine, T., Mann, H., Marie, I., Merola, J., Olesinska, M., Olsen, N., Pipitone, N., Ramchandren, S., Rutkove, S., Saketkoo, L. A., Schiffenbauer, A., Selva-O'Callaghan, A., Shinjo, S. K., Shupak, R., Swierkocka, K., de Visser, M., Wanschitz, J., Werth, V. P., Whitt, I., Ytterberg, S., Avcin, T., Becker, M., Beresford, M. W., Dressler, F., Dvergsten, J., Ferriani, V. P. L., Flato, B., Gerloni, V., Henrickson, M., Hinze, C., Hoeltzel, M., BUNES IBARRA, MIGUEL ANGEL, Ilowite, N., Imundo, L., Kingsbury, D., Magnusson, B., Maguiness, S., Mathiesen, P., Mccann, L., Nielsen, S., de Oliveira, S. K. F., Passo, M., Rabinovich, E., Rivas-Chacon, R., Robinson, A. B., Rouster-Stevens, K., Russo, R., Rutkowska-Sak, L., Sallum, A., Sanner, H., Schmeling, H., Selcen, D., Shaham, B., Spencer, C. H., Sundel, R., Tardieu, M., Thatayatikom, A., Wahezi, D., and Zulian, F.

Subjects
medicine.medical_specialty, dermatomyositis, Hybrid measure, 1000Minds software, Minimal Clinically Important Difference, response criteria, Klinikai orvostudományok, Logistic regression, Polymyositis, Dermatomyositis, polymyositis, 03 medical and health sciences, 0302 clinical medicine, Rheumatology, Response criteria, medicine, Humans, Pharmacology (medical), Conjoint analysis, outcome assessment, Juvenile dermatomyositis, Myositis, 030203 arthritis & rheumatology, hybrid measure, juvenile dermatomyositis, business.industry, Minimal clinically important difference, Outcome assessment, Area Under Curve, Logistic Models, Treatment Outcome, Orvostudományok, Clinical Science, medicine.disease, Adult dermatomyositis, Clinical trial, Physical therapy, conjoint analysis, business, 030217 neurology & neurosurgery
Abstract

Objective: The objective was to describe the methodology used to develop new response criteria for adult DM/PM and JDM.Methods: Patient profiles from prospective natural history data and clinical trials were rated by myositis specialists to develop consensus gold-standard ratings of minimal, moderate and major improvement. Experts completed a survey regarding clinically meaningful improvement in the core set measures (CSM) and a conjoint-analysis survey (using 1000Minds software) to derive relative weights of CSM and candidate definitions. Six types of candidate definitions for response criteria were derived using survey results, logistic regression, conjoint analysis, application of conjoint-analysis weights to CSM and published definitions. Sensitivity, specificity and area under the curve were defined for candidate criteria using consensus patient profile data, and selected definitions were validated using clinical trial data.Results: Myositis specialists defined the degree of clinically meaningful improvement in CSM for minimal, moderate and major improvement. The conjoint-analysis survey established the relative weights of CSM, with muscle strength and Physician Global Activity as most important. Many candidate definitions showed excellent sensitivity, specificity and area under the curve in the consensus profiles. Trial validation showed that a number of candidate criteria differentiated between treatment groups. Top candidate criteria definitions were presented at the consensus conference.Conclusion: Consensus methodology, with definitions tested on patient profiles and validated using clinical trials, led to 18 definitions for adult PM/DM and 14 for JDM as excellent candidates for consideration in the final consensus on new response criteria for myositis.

Published
2017
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43. Evaluation of risk factors and biomarkers related to arterial and venous thrombotic events in idiopathic inflammatory myopathies.

Author

Notarnicola, A, Barsotti, S, Näsman, L, Tang, Q, Holmqvist, M, Lundberg, IE, Antovic, A, and Lundberg, I E

Subjects
PHOSPHOLIPID antibodies, MYOSITIS, RISK assessment, ESSENTIAL hypertension, MUSCLE diseases, OLDER people, BIOMARKERS
Abstract

Objectives: This study aimed to assess the contribution of traditional/disease-related risk factors and biomarkers linked to arterial and venous thrombotic events (TEs) in patients with idiopathic inflammatory myopathies (IIMs).Method: The occurrence of arterial and/or venous TEs at the time of or after IIM diagnosis was retrospectively evaluated in a cohort of 253 patients with IIMs, resulting in a final population of 246 IIM patients, 51 with reported TE (cases) and 195 without a history of TE (comparators). Information on disease characteristics and traditional risk factors for arterial and venous TE (essential hypertension, diabetes, dyslipidaemia, smoking, malignancy) was retrieved. Serum levels of anti-phospholipid antibodies (aPLs) and adhesion molecules were analysed at the time of IIM diagnosis and at the time of the TE in cases.Results: One in five IIM patients (21%) had experienced a TE, arterial TE in 22 and venous TE in 29 patients, with a peak prevalence within 5 years after diagnosis. Among traditional/disease-related risk factors, only older age was associated with both arterial and venous TEs, after adjusting for other covariates. Low serum levels of e-selectin were associated with higher odds of developing a TE, without specific association with either arterial or venous TEs. Only 6% of IIM patients had aPLs, with no significant difference between cases and comparators.Conclusions: An increased risk of both venous and arterial TEs should be considered in IIM patients, particularly close to diagnosis and in elderly people. Low serum levels of e-selectin may predict TE in IIM patients, but the underlying biological mechanism is not known. [ABSTRACT FROM AUTHOR]

Published
2021
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44. Educational needs and preferences of young European clinicians and physician researchers working in the field of rheumatology

Author

Beyer, C, Ramiro, S, Sivera, F, Mandl, P, Machado, P, Ospelt, C, Molto, A, Radner, H, Iagnocco, A, Bijlsma, J, Lundberg, I, Grigoriou, A, Fanouriakis, A, Hueber, A, Maurer, B, Scire, C, Gaujoux-Viala, C, Karpec, D, Van Der Woude, D, Rimar, D, Deseatnicova, E, Nikiphorou, E, De Langhe, E, Haavardsholm, E, Starodubtseva, I, Nagy, G, Biliavska, I, Sznajd, J, Karlsson, J, Zepa, J, Cassar, K, Senolt, L, Ghukasyan, L, Baresic, M, Frank-Bertoncelj, M, Ziade, N, Gobejishvili, N, Ostojic, P, Ostgard, R, Dobrota, R, Shumnalieva, R, Talli, S, Panchal, S, Volpinari, S, Rannio, T, Romao, V, Beyer C., Ramiro S., Sivera F., Mandl P., MacHado P. M., Ospelt C., Molto A., Radner H., Iagnocco A., Bijlsma J. W., Lundberg I. E., Grigoriou A., Fanouriakis A., Hueber A., Maurer B., Scire C. A., Gaujoux-Viala C., Karpec D., Van Der Woude D., Rimar D., Deseatnicova E., Nikiphorou E., De Langhe E., Haavardsholm E. A., Starodubtseva I., Nagy G., Biliavska I. V., Sznajd J., Karlsson J. A., Zepa J., Cassar K., Senolt L., Ghukasyan L., Baresic M., Frank-Bertoncelj M., Ziade N., Gobejishvili N., Ostojic P., Ostgard R., Dobrota R., Shumnalieva R., Talli S., Panchal S., Volpinari S., Rannio T., Romao V. C., Beyer, C, Ramiro, S, Sivera, F, Mandl, P, Machado, P, Ospelt, C, Molto, A, Radner, H, Iagnocco, A, Bijlsma, J, Lundberg, I, Grigoriou, A, Fanouriakis, A, Hueber, A, Maurer, B, Scire, C, Gaujoux-Viala, C, Karpec, D, Van Der Woude, D, Rimar, D, Deseatnicova, E, Nikiphorou, E, De Langhe, E, Haavardsholm, E, Starodubtseva, I, Nagy, G, Biliavska, I, Sznajd, J, Karlsson, J, Zepa, J, Cassar, K, Senolt, L, Ghukasyan, L, Baresic, M, Frank-Bertoncelj, M, Ziade, N, Gobejishvili, N, Ostojic, P, Ostgard, R, Dobrota, R, Shumnalieva, R, Talli, S, Panchal, S, Volpinari, S, Rannio, T, Romao, V, Beyer C., Ramiro S., Sivera F., Mandl P., MacHado P. M., Ospelt C., Molto A., Radner H., Iagnocco A., Bijlsma J. W., Lundberg I. E., Grigoriou A., Fanouriakis A., Hueber A., Maurer B., Scire C. A., Gaujoux-Viala C., Karpec D., Van Der Woude D., Rimar D., Deseatnicova E., Nikiphorou E., De Langhe E., Haavardsholm E. A., Starodubtseva I., Nagy G., Biliavska I. V., Sznajd J., Karlsson J. A., Zepa J., Cassar K., Senolt L., Ghukasyan L., Baresic M., Frank-Bertoncelj M., Ziade N., Gobejishvili N., Ostojic P., Ostgard R., Dobrota R., Shumnalieva R., Talli S., Panchal S., Volpinari S., Rannio T., and Romao V. C.

Abstract

Objectives: To understand the educational needs and preferences of young clinicians and physician researchers in the field of rheumatology in Europe. Methods: An international online survey was performed as a joint venture of ESCET and EMEUNET. The survey assessed the acceptance of and the access to the current European League Against Rheumatism (EULAR) educational portfolio, as well as the unmet educational needs and learning preferences among individuals below the age of 40 years working in rheumatology in Europe. Results: Among 568 European clinicians and physician researchers, 65% indicated that the existing EULAR educational portfolio adequately covers their educational needs. Within the EULAR portfolio, the online course on rheumatic diseases and the postgraduate course were the most appreciated. Participants were very much in favour of new educational courses on imaging techniques, and 63% of participants indicated a particular interest in musculoskeletal ultrasound. A strong interest in refresher (60%) and general review (55%) courses was observed. Lack of funding was considered the major obstacle to participating in existing EULAR programmes. Finally, participants showed diverse preferences regarding learning modalities with common interests in live courses and conferences. Conclusions: EULAR's training opportunities are well appreciated among young clinicians and physician researchers in rheumatology. The results from this survey will help to develop EULAR's future educational portfolio.

Published
2016

46. Enhanced ventricular-arterial coupling during a 2-year physical activity programme in patients with rheumatoid arthritis : a prospective substudy of the physical activity in rheumatoid arthritis 2010 trial.

Author

Sarajlic, P, Fridén, C, Lund, L H, Manouras, A, Venkateshvaran, A, Larsson, Susanna C., Nordgren, B, Opava, C H, Lundberg, I E, Bäck, M, Sarajlic, P, Fridén, C, Lund, L H, Manouras, A, Venkateshvaran, A, Larsson, Susanna C., Nordgren, B, Opava, C H, Lundberg, I E, and Bäck, M

Abstract

OBJECTIVE: To establish how guided physical activity in patients with rheumatoid arthritis (RA) without known cardiovascular disease affected vascular and cardiac function, and how these two entities were prospectively interconnected in this patient group. METHODS: Prospective substudy of 29 participants in the Physical Activity in RA (PARA) 2010 trial. All subjects were examined at baseline, at year 1 and 2 with measures of pulse wave velocity and arterial augmentation index, as well as echocardiographic evaluation of diastolic parameters and ventricular-arterial coupling. Muscle strength and aerobic exercise capacity were assessed at baseline and yearly. All participants performed physiotherapist-guided aerobic and muscle strength exercise during 2 years and were reminded through SMS to report physical activity progress. RESULTS: This cohort of patients with RA exhibited increased vascular stiffness despite normal blood pressure. At baseline, lower muscle strength was associated with increased vascular stiffness (β = 0.68; P = 0.004), whereas lower aerobic working capacity was associated with left ventricular diastolic dysfunction (β = 0.85; P = 0.03). There was a significant positive correlation between vascular stiffness and diastolic dysfunction at baseline (R2 = 0.64) and for the changes in those parameters observed during 2 years of guided physical activity. Finally, a significant improvement in ventricular-arterial coupling was observed after exercise (P < 0.001). CONCLUSION: These results indicate that although differentially associated with physical capacity parameters, improved vascular stiffness and improved diastolic dysfunction are interrelated, and that an optimization of the ventricular-arterial coupling may contribute to the beneficial effects of physical activity in patients with RA.

Published
2018
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47. Molecular mimicry between the autoantigen Anoctamin 2 and Epstein Barr virus nuclear antigen 1 associates with increased risk for multiple sclerosis

Author

Olsson, T., Huang, J., Hellström, Cecilia, Tengvall, K., Kammer, P., Bistrom, M., Bomfim, I. Lima, Stridh, P., Butt, J., Nicole, B., Michel, A., Ayoglu, Burcu, Lundberg, K., Lundberg, I., Olafsson, S., Jonsdottir, I., Stefansson, K., Dilthey, A., Hillert, J., Alfredsson, L., Sundstrom, P., Nilsson, Peter, Waterboer, T., Kockum, I., Olsson, T., Huang, J., Hellström, Cecilia, Tengvall, K., Kammer, P., Bistrom, M., Bomfim, I. Lima, Stridh, P., Butt, J., Nicole, B., Michel, A., Ayoglu, Burcu, Lundberg, K., Lundberg, I., Olafsson, S., Jonsdottir, I., Stefansson, K., Dilthey, A., Hillert, J., Alfredsson, L., Sundstrom, P., Nilsson, Peter, Waterboer, T., and Kockum, I.

Abstract

QC 20181121

Published
2018

50. Pulse versus daily oral cyclophosphamide for induction of remission in ANCA-associated vasculitis: long-term follow-up

Author

Harper, L1, Morgan, Md, Walsh, M, Hoglund, P, Westman, K, Flossmann, O, Tesar, V, Vanhille, P, de Groot, K, Luqmani, R, Flores Suarez LF, Watts, R, Pusey, C, Bruchfeld, A, Rasmussen, N, Blockmans, D, Savage, Co, Jayne, D, EUVAS i. n. v. e. s. t. i. g. a. t. o. r. s. Abramowicz, D, Wissing, M, Madhoun, P, Stolear, J, Ekstrand, A, Chabova, V, Rychlik, I, Bataille, P, Puechal, X, Leblau, J, Esnault, Vl, Gross, Wl, Weidner, S, Rupprecht, H, Schneider, M, Specker, C, Schmitt, W, van der Woude, F, Vischedyck, M, Feighery, C, Garibotto, Giacomo, Sinico, Ra, Santostefano, M, Dadoniene, J, Hagen, Ec, Oudejans, I, Verburgh, C, Ballarin, J, Mirapeix, E, Valles, M, Pettersson, E, Eriksson, P, Selga, D, Segelmark, M, Sterner, G, Lundberg, I, Svenungsson, E, Chizzolini, C, Adu, D, de Souza, R., and Chizzolini, Carlo

Subjects
Male, Time Factors, Administration, Oral, Kidney, law.invention, Cyclophosphamide/administration & dosage, Randomized controlled trial, law, Risk Factors, Secondary Prevention, Immunology and Allergy, ddc:616, Aged, 80 and over, ANCA, Remission Induction, Middle Aged, Female, Vasculitis, Immunosuppressive Agents, medicine.drug, Systemic vasculitis, Adult, medicine.medical_specialty, Cyclophosphamide, Adolescent, Kidney/physiology, Immunology, General Biochemistry, Genetics and Molecular Biology, Antibodies, Antineutrophil Cytoplasmic, Young Adult, Rheumatology, Internal medicine, medicine, Kidney Function, Humans, Adverse effect, Immunosuppressive Agents/administration & dosage, Aged, Retrospective Studies, business.industry, Vasculitis/drug therapy/immunology/mortality, Antibodies, Antineutrophil Cytoplasmic/immunology, Retrospective cohort study, medicine.disease, Surgery, Regimen, Pulse Therapy, Drug, business, Follow-Up Studies
Abstract

INTRODUCTION: The previously reported randomised controlled trial of a consensus regimen of pulse cyclophosphamide suggested that it was as effective as a daily oral (DO) cyclophosphamide for remission induction of antineutrophil cytoplasm autoantibodies-associated systemic vasculitis when both were combined with the same glucocorticoid protocol (CYCLOPS study (Randomised trial of daily oral versus pulse Cyclophosphamide as therapy for ANCA-associated Systemic Vasculitis published de groot K, harper L et al Ann Int Med 2009)). The study had limited power to detect a difference in relapse. This study describes the long-term outcomes of patients in the CYCLOPS study. METHODS: Long-term outcomes were ascertained retrospectively from 148 patients previously recruited to the CYCLOPS Trial. Data on survival, relapse, immunosuppressive treatment, cancer incidence, bone fractures, thromboembolic disease and cardiovascular morbidity were collected from physician records retrospectively. All patients were analysed according to the group to which they were randomised. RESULTS: Median duration of follow-up was 4.3 years (IQR, 2.95-5.44 years). There was no difference in survival between the two limbs (p=0.92). Fifteen (20.8%) DO and 30 (39.5%) pulse patients had at least one relapse. The risk of relapse was significantly lower in the DO limb than the pulse limb (HR=0.50, 95% CI 0.26 to 0.93; p=0.029). Despite the increased risk of relapse in pulse-treated patients, there was no difference in renal function at study end (p=0.82). There were no differences in adverse events between the treatment limbs. DISCUSSION: Pulse cyclophosphamide is associated with a higher relapse risk than DO cyclophosphamide. However, this is not associated with increased mortality or long-term morbidity. Although the study was retrospective, data was returned in 90% of patients from the original trial.

Published
2016

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