Healthcare utilization and unmet needs of patients with antisynthetase syndrome: An international patient survey.

Jul 17, 2023

Experts: M Weiss,M T Holzer,F Muehlensiepen,Y Ignatyev,C Fiehn,J Bauhammer,J Schmidt,S Schlüter,A Dihkan,D Scheibner,U Schneider,L Valor-Mendez,G Corte,L Gupta,H Chinoy,I Lundberg,L Cavagna,J H W Distler,G Schett,J Knitza

Antisynthease syndrome (ASSD) is a rare, complex and understudied autoimmune disease. Internet-based studies can overcome barriers of traditional on-site research and are therefore very appealing for rare diseases. The aim of this study was to investigate patient-reported symptoms, diagnostic delay, symptoms, medical care, health status, working status, disease knowledge and willingness to participate in research of ASSD patients by conducting an international web-based survey. The multilingual questionnaire was created by an international group of rheumatologists and patients and distributed online. 236 participants from 22 countries completed the survey. 184/236 (78.0%) were female, mean age (SD) was 49.6 years (11.3) and most common antisynthetase antibody was Jo-1 (169/236, 71.6%). 79/236 (33.5%) reported to work full-time. Median diagnostic delay was one year. The most common symptom at disease onset was fatigue 159/236 (67.4%), followed by myalgia 130/236 (55.1%). The complete triad of myositis, arthritis and lung involvement verified by a clinician was present in 42/236 (17.8%) at disease onset and in 88/236 (37.3%) during the disease course. 36/236 (15.3%) reported to have been diagnosed with fibromyalgia and 40/236 (16.3%) with depression. The most reported immunosuppressive treatments were oral corticosteroids 179/236 (75.9%), followed by rituximab 85/236 (36.0%). 73/236 (30.9%) had received physiotherapy treatment. 71/236 (30.1%) reported to know useful online information sources related to ASSD. 223/236 (94.5%) were willing to share health data for research purposes once a year. Our results reiterate that internet-based research is invaluable for cooperating with patients to foster knowledge in rare diseases.
© 2023. The Author(s).

Author

admin

View all posts

ABOUT THE EXPERTS

M Weiss,M T Holzer,F Muehlensiepen,Y Ignatyev,C Fiehn,J Bauhammer,J Schmidt,S Schlüter,A Dihkan,D Scheibner,U Schneider,L Valor-Mendez,G Corte,L Gupta,H Chinoy,I Lundberg,L Cavagna,J H W Distler,G Schett,J Knitza

M Weiss

Department of Internal Medicine 3, Friedrich-Alexander-University Erlangen-Nürnberg and Universitätsklinikum Erlangen, Erlangen, Germany.

Deutsches Zentrum Immuntherapie (DZI), Friedrich-Alexander-University, Erlangen, Germany.

M T Holzer

III. Department of Internal Medicine, University Medical Center Hamburg-Eppendorf, Hamburg, Germany. m.holzer@uke.de.

F Muehlensiepen

Faculty of Health Sciences, Center for Health Services Research, Brandenburg Medical School Theodor Fontane, Rüdersdorf, Germany.

Y Ignatyev

Faculty of Health Sciences, Center for Health Services Research, Brandenburg Medical School Theodor Fontane, Rüdersdorf, Germany.

C Fiehn

Praxis für Rheumatologie, Klinische Immunologie, Medical Center, Baden-Baden, Germany.

J Bauhammer

Praxis für Rheumatologie, Klinische Immunologie, Medical Center, Baden-Baden, Germany.

J Schmidt

Faculty of Health Sciences Brandenburg, Brandenburg Medical School Theodor Fontane, Rüdersdorf bei Berlin, Germany.

Department of Neurology and Pain Treatment, Center for Translational Medicine, Neuromuscular Center, Immanuel Klinik Rüdersdorf, University Hospital of the Brandenburg Medical School Theodor Fontane, Rüdersdorf bei Berlin, Germany.

Department of Neurology, Neuromuscular Center, University Medical Center, Göttingen, Germany.

S Schlüter

Myositis-Gruppe, Deutsche Gesellschaft Für Muskelkranke, Freiburg, Germany.

A Dihkan

The Swedish Working Group for Myositis, The Swedish Rheumatism Association, Stockholm, Sweden.

D Scheibner

Myositis-Gruppe, Deutsche Gesellschaft Für Muskelkranke, Freiburg, Germany.

U Schneider

Department of Rheumatology, Charité – Universitätsmedizin Berlin, Corporate Member of Freie Universität Berlin and Humboldt-Universität zu Berlin, Berlin, Germany.

L Valor-Mendez

Department of Internal Medicine 3, Friedrich-Alexander-University Erlangen-Nürnberg and Universitätsklinikum Erlangen, Erlangen, Germany.

Deutsches Zentrum Immuntherapie (DZI), Friedrich-Alexander-University, Erlangen, Germany.

G Corte

Department of Internal Medicine 3, Friedrich-Alexander-University Erlangen-Nürnberg and Universitätsklinikum Erlangen, Erlangen, Germany.

Deutsches Zentrum Immuntherapie (DZI), Friedrich-Alexander-University, Erlangen, Germany.

L Gupta

Department of Rheumatology, Royal Wolverhampton Hospitals NHS Trust, Wolverhampton, UK.

Division of Musculoskeletal and Dermatological Sciences, School of Biological Sciences, Centre for Musculoskeletal Research, The University of Manchester, Manchester, UK.

Department of Rheumatology, City Hospital, Sandwell and West Birmingham Hospitals NHS Trust, Birmingham, UK.

H Chinoy

Department of Rheumatology, Salford Royal Hospital, Northern Care Alliance NHS Foundation Trust, Manchester Academic Health Science Centre, Salford, UK.

Division of Musculoskeletal and Dermatological Sciences, Faculty of Biology, Medicine and Health, The University of Manchester, Manchester, UK.

I Lundberg

Division of Rheumatology, Department of Medicine, Karolinska Institutet, Karolinska University Hospital, Solna, Stockholm, Sweden.

Department of Gastroenterology, Dermatology and Rheumatology, Karolinska University Hospital, Stockholm, Sweden.

L Cavagna

Rheumatology Division, Fondazione Istituti di Ricovero e Cura a Carattere Scientifico (IRCCS) Policlinico San Matteo, Pavia, Italy.

J H W Distler

Department of Internal Medicine 3, Friedrich-Alexander-University Erlangen-Nürnberg and Universitätsklinikum Erlangen, Erlangen, Germany.

Deutsches Zentrum Immuntherapie (DZI), Friedrich-Alexander-University, Erlangen, Germany.

G Schett

Department of Internal Medicine 3, Friedrich-Alexander-University Erlangen-Nürnberg and Universitätsklinikum Erlangen, Erlangen, Germany.

Deutsches Zentrum Immuntherapie (DZI), Friedrich-Alexander-University, Erlangen, Germany.

J Knitza

Department of Internal Medicine 3, Friedrich-Alexander-University Erlangen-Nürnberg and Universitätsklinikum Erlangen, Erlangen, Germany.

Deutsches Zentrum Immuntherapie (DZI), Friedrich-Alexander-University, Erlangen, Germany.