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

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

doi:10.1007/s00296-023-05372-9

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

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

Division of Musculoskeletal & Dermatological Sciences (L5)

Research output: Contribution to journal › Article › peer-review

Abstract

Introduction: 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.
Objective: 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.
Methods: The multilingual questionnaire was created by an international group of rheumatologists and patients and distributed online.
Results: 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.
Conclusion: Our results reiterate that internet-based research is invaluable for cooperating with patients to foster knowledge in rare diseases.

Original language	English
Journal	Rheumatology International
Early online date	15 Jul 2023
DOIs	https://doi.org/10.1007/s00296-023-05372-9
Publication status	E-pub ahead of print - 15 Jul 2023

Keywords

myositis
autoantibodies
health services research
therapeutics
diagnosis

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10.1007/s00296-023-05372-9Licence: CC BY

NIHR Manchester Biomedical Research Centre
Bruce, I. (PI), Lord, G. (CoI), Lennon, R. (CoI), Black, G. (CoI), Wedge, D. (CoI), Morris, A. (CoI), Hussell, T. (CoI), Sharrocks, A. (CoI), Stivaros, S. (CoI), Buch, M. (CoI), Gough, J. (CoI), Kostarelos, K. (CoI), Thistlethwaite, F. (CoI), Kadler, K. (CoI), Barton, A. (CoI), Hyrich, K. (CoI), Mcbeth, J. (CoI), O'Neill, T. (CoI), Vestbo, J. (CoI), Simpson, A. (CoI), Singh, S. (CoI), Smith, J. (CoI), Felton, T. (CoI), Murray, C. (CoI), Griffiths, C. (CoI), Cullum, N. (CoI), Rhodes, L. (CoI), Warren, R. (CoI), Paus, R. (CoI), Dumville, J. (CoI), Viros Usandizaga, A. (CoI), Keavney, B. (CoI), Tomaszewski, M. (CoI), Allan, S. (CoI), Body, R. (CoI), Cartwright, E. (CoI), Heagerty, A. (CoI), Kalra, P. (CoI), Miller, C. (CoI), Rutter, M. (CoI), Smith, C. (CoI), Trafford, A. (CoI), Evans, D. (CoI), Crosbie, E. (CoI), Crosbie, P. (CoI), Harvie, M. (CoI), Howell, S. (CoI), Renehan, A. (CoI), Dive, C. (CoI), Blackhall, F. (CoI), Landers, D. (CoI), Krebs, M. (CoI), Cook, N. (CoI), Clarke, R. (CoI), Taylor, S. (CoI), Jorgensen, C. (CoI), Lorigan, P. (CoI), Jayson, G. (CoI), Valle, J. (CoI), Mccabe, M. (CoI), Armstrong, A. (CoI), Freitas, A. (CoI), Illidge, T. (CoI), Choudhury, A. (CoI), Hoskin, P. (CoI), West, C. (CoI), Van Herk, M. (CoI), Faivre-Finn, C. (CoI), Bristow, R. (CoI), Kirkby, K. (CoI), Birtle, A. (CoI), Mackay, R. (CoI), Radford, J. (CoI), Linton, K. (CoI), Higham, C. (CoI), Munro, K. (CoI), Plack, C. (CoI), Arden Armitage, C. (CoI), Bruce, I. (CoI), Moore, D. (CoI), Saunders, G. (CoI), Stone, M. (CoI), Haddock, G. (CoI), Lewis, S. (CoI), Elliott, R. (CoI), Green, J. (CoI), Lovell, K. (CoI), Morrison, A. (CoI), Shaw, J. (CoI), Bucci, S. (CoI), Ainsworth, J. (CoI), Webb, R. (CoI), Newman, W. (CoI), Banka, S. (CoI), Clayton-Smith, J. (CoI), Payne, K. (CoI), Moldovan, R. (CoI), Wynn, R. (CoI) & Jones, S. (CoI)
1/12/22 → 30/11/27
Project: Research
MMRG: Manchester Myositis Research Group
Chinoy, H. (PI), Lamb, J. (PI), Ollier, W. (PI), Rothwell, S. (CoI), Lilleker, J. (CoI), Oldroyd, A. (PGR student), Snedden, A. (PGR student), Platt, H. (Support team) & New, P. (Support team)
1/01/10 → …
Project: Research

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Chinoy, H., Dihkan, A., Schlüter, S., Schmidt, J., Bauhammer, J., Fiehn, C., Ignatyev, Y., Muehlensiepen, F., Holzer, M. T., Weiss, M., Scheibner, D., Schneider, U., Valor Mendez, L., Corte, G., Gupta, L., Lundberg, I., Cavagna, L., Distler, J. H. W., Schett, G., & Knitza, J. (2023). Healthcare utilization and unmet needs of patients with antisynthetase syndrome: An international patient survey. Rheumatology International. Advance online publication. https://doi.org/10.1007/s00296-023-05372-9

@article{04728ca9f1ec4f8fbd5ce91db2a2af66,

title = "Healthcare utilization and unmet needs of patients with antisynthetase syndrome: An international patient survey",

abstract = "Introduction: 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. Objective: 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. Methods: The multilingual questionnaire was created by an international group of rheumatologists and patients and distributed online. Results: 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. Conclusion: Our results reiterate that internet-based research is invaluable for cooperating with patients to foster knowledge in rare diseases. ",

keywords = "myositis, autoantibodies, health services research, therapeutics, diagnosis",

author = "Hector Chinoy and A. Dihkan and S. Schl{\"u}ter and J. Schmidt and J. Bauhammer and C. Fiehn and Y. Ignatyev and F. Muehlensiepen and M.T. Holzer and M. Weiss and D. Scheibner and U. Schneider and \{Valor Mendez\}, L. and G. Corte and Latika Gupta and I. Lundberg and L. Cavagna and J.H.W. Distler and G. Schett and J Knitza",

year = "2023",

month = jul,

day = "15",

doi = "10.1007/s00296-023-05372-9",

language = "English",

journal = "Rheumatology International",

issn = "0172-8172",

publisher = "Springer Nature",

}

Chinoy, H, Dihkan, A, Schlüter, S, Schmidt, J, Bauhammer, J, Fiehn, C, Ignatyev, Y, Muehlensiepen, F, Holzer, MT, Weiss, M, Scheibner, D, Schneider, U, Valor Mendez, L, Corte, G, Gupta, L, Lundberg, I, Cavagna, L, Distler, JHW, Schett, G & Knitza, J 2023, 'Healthcare utilization and unmet needs of patients with antisynthetase syndrome: An international patient survey', Rheumatology International. https://doi.org/10.1007/s00296-023-05372-9

TY - JOUR

T1 - Healthcare utilization and unmet needs of patients with antisynthetase syndrome: An international patient survey

AU - Chinoy, Hector

AU - Dihkan, A.

AU - Schlüter, S.

AU - Schmidt, J.

AU - Bauhammer, J.

AU - Fiehn, C.

AU - Ignatyev, Y.

AU - Muehlensiepen, F.

AU - Holzer, M.T.

AU - Weiss, M.

AU - Scheibner, D.

AU - Schneider, U.

AU - Valor Mendez, L.

AU - Corte, G.

AU - Gupta, Latika

AU - Lundberg, I.

AU - Cavagna, L.

AU - Distler, J.H.W.

AU - Schett, G.

AU - Knitza, J

PY - 2023/7/15

Y1 - 2023/7/15

N2 - Introduction: 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. Objective: 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. Methods: The multilingual questionnaire was created by an international group of rheumatologists and patients and distributed online. Results: 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. Conclusion: Our results reiterate that internet-based research is invaluable for cooperating with patients to foster knowledge in rare diseases.

AB - Introduction: 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. Objective: 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. Methods: The multilingual questionnaire was created by an international group of rheumatologists and patients and distributed online. Results: 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. Conclusion: Our results reiterate that internet-based research is invaluable for cooperating with patients to foster knowledge in rare diseases.

KW - myositis

KW - autoantibodies

KW - health services research

KW - therapeutics

KW - diagnosis

U2 - 10.1007/s00296-023-05372-9

DO - 10.1007/s00296-023-05372-9

M3 - Article

SN - 0172-8172

JO - Rheumatology International

JF - Rheumatology International

ER -

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

Abstract

Keywords

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NIHR Manchester Biomedical Research Centre

MMRG: Manchester Myositis Research Group

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