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Rheumatol Adv Pract
Rheumatol Adv Pract
rheumap
Rheumatology Advances in Practice
2514-1775
Oxford University Press

10.1093/rap/rkae098
rkae098
Letter to the Editor
Letter to the Editor (Matters arising from published papers)
AcademicSubjects/MED00010
Comment on: Impaired health-related quality of life in idiopathic inflammatory myopathies: a cross-sectional analysis from the COVAD-2 e-survey: Reply
https://orcid.org/0000-0003-3590-1637
Yoshida Akira Department of Allergy and Rheumatology, Nippon Medical School Graduate School of Medicine, Tokyo, Japan

https://orcid.org/0000-0001-8352-6136
Kuwana Masataka Department of Allergy and Rheumatology, Nippon Medical School Graduate School of Medicine, Tokyo, Japan

Agarwal Vikas Department of Clinical Immunology and Rheumatology, Sanjay Gandhi Postgraduate Institute of Medical Sciences, Lucknow, India

https://orcid.org/0000-0003-2753-2990
Gupta Latika Department of Rheumatology, Royal Wolverhampton Hospitals NHS Trust, Wolverhampton, UK
Division of Musculoskeletal and Dermatological Sciences, Centre for Musculoskeletal Research, School of Biological Sciences, University of Manchester, Manchester, UK

CoVAD Study Group authors Chinoy Hector
Nikiphorou Elena
Tan Ai Lyn
Nune Arvind
Lilleker James B
Pauling John D
Wincup Chris
Gasparyan Armen Yuri
Sen Parikshit
Joshi Mrudula
Agarwal Vishwesh
Kadam Esha
Barman Bhupen
Singh Yogesh Preet
Ranjan Rajiv
Jain Avinash
Pandya Sapan C
Pilania Rakesh Kumar
Sharma Aman
Manesh Manoj M
Gupta Vikas
Kavadichanda Chengappa G
Patro Pradeepta Sekhar
Ajmani Sajal
Phatak Sanat
Goswami Rudra Prosad
Chowdhury Abhra Chandra
Mathew Ashish Jacob
Shenoy Padnamabha
Asranna Ajay
Bommakanti Keerthi Talari
Shukla Anuj
Pande Arunkumar R
Gaur Prithvi Sanjeevkumar
Mamadapur Mahabaleshwar
Ghodke Akanksha
Chandwar Kunal
Salim Babur
Fazal Zoha Zahid
Kardeş Sinan
Cansu Döndü Üsküdar
Yıldırım Reşit
Makol Ashima
Chatterjee Tulika
Patel Aarat
Giannini Margherita
Maurier François
Campagne Julien
Meyer Alain
Cavagna Lorenzo
Del Papa Nicoletta
Sambataro Gianluca
Fabiola Atzeni
Govoni Marcello
Parisi Simone
Bocci Elena Bartoloni
Sebastiani Gian Domenico
Fusaro Enrico
Sebastiani Marco
Quartuccio Luca
Franceschini Franco
Sainaghi Pier Paolo
Orsolini Giovanni
De Angelis Rossella
Danielli Maria Giovanna
Venerito Vincenzo
Grignaschi Silvia
Giollo Alessandro
Andreoli Laura
Lini Daniele
Alunno Alessia
Iannone Florenzo
Fornaro Marco
Traboco Lisa S
Shaharir Syahrul Sazliyana
Wibowo Suryo Anggoro Kusumo
Gracia-Ramos Abraham Edgar
Saavedra Miguel A
Tehozol Erick Adrian Zamora
Serrano Jorge Rojas
La Torre Ignacio García-De
Colunga‑Pedraza Iris J
Merayo-Chalico Javier
Aranega Raquel
Loarce-Martos Jesús
Prieto-González Sergio
Shinjo Samuel Katsuyuki
Hoff Leonardo Santos
Nakashima Ran
Sato Shinji
Kimura Naoki
Kaneko Yuko
Gono Takahisa
Parodis Ioannis
Milchert Marcin
Distler Oliver
Knitza Johannes
Tomaras Stylianos
Proft Fabian Nikolai
Holzer Marie-Therese
Schreiber Karen
Gromova Margarita Aleksandrovna
Aharonov Or
Nagy-Vincze Melinda
Griger Zoltán
Ziade Nelly
Hmamouchi Ihsane
El bouchti Imane
Baba Zineb
Dey Dzifa
Ima-Edomwonyi Uyi
Dedeke Ibukunoluwa
Airenakho Emorinken
Madu Nwankwo Henry
Yerima Abubakar
Olaosebikan Hakeem
Okwara Celestine Chibuzo
Becky A
Koussougbo Ouma Devi
Palalane Elisa
Day Jessica
Langguth Daman
Limaye Vidya
Needham Merrilee
Srivastav Nilesh
Hudson Marie
Landon-Cardinal Océane
Velikova Tsvetelina
Shumnalieva Russka
Gutiérrez Carlos Enrique Toro
Caballero-Uribe Carlo Vinicio
Zuleta Wilmer Gerardo Rojas
Arbeláez Álvaro
Cajas Javier
Silva José António Pereira
Fonseca João Eurico
Zimba Olena
So Ho
Ugarte-Gil Manuel Francisco
Chinchay Lyn
Bernaola José Proaño
Pimentel Victorio
Saha Sreoshy
Hasan A T M Tanveer
Vaidya Binit
Gheita Tamer A
Fathi Hanan Mohamed
Mohammed Reem Hamdy A
Chen Yi-Ming
Harifi Ghita
El Kibbi Lina
Halabi Hussein
Goo Phonpen Akawatcharangura
Katchamart Wanruchada
Fuentes-Silva Yurilís
Cabriza Karoll
Losanto Jonathan
Colaman Nelly
Cachafeiro-Vilar Antonio
Bautista Generoso Guerra
Ho Enrique Julio Giraldo
Nunez Lilith Stange
Cristian Vergara M
Báez Jossiell Then
Alonzo Hugo
Pastelin Carlos Benito Santiago
Salinas Rodrigo García
Obiols Alejandro Quiñónez
Chávez Nilmo
Ordóñez Andrea Bran
Llerena Gil Alberto Reyes
Sierra-Zorita Radames
Arrieta Dina
Hidalgo Eduardo Romero
Saenz Ricardo
Idania Escalante M
Calapaqui Wendy
Quezada Ivonne
Arredondo Gabriela

Correspondence to: Latika Gupta, Department of Rheumatology, Royal Wolverhampton Hospitals NHS Trust, Wolverhampton WV10 0QP, UK. E-mail: drlatikagupta@gmail.com
‡ The complete list of authors of the COVAD Study Group as well as their affiliations are provided in the Supplementary Material, available at Rheumatology Advances in Practice online.

2024
14 8 2024
14 8 2024
8 3 rkae09812 8 2024
04 9 2024
© The Author(s) 2024. Published by Oxford University Press on behalf of the British Society for Rheumatology.
2024
https://creativecommons.org/licenses/by-nc/4.0/ This is an Open Access article distributed under the terms of the Creative Commons Attribution-NonCommercial License (https://creativecommons.org/licenses/by-nc/4.0/), which permits non-commercial re-use, distribution, and reproduction in any medium, provided the original work is properly cited. For commercial re-use, please contact journals.permissions@oup.com
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pmc Dear Editor, We thank Finsterer for the comments on our article [1], in which we investigated health-related quality of life (HRQoL) in patients with idiopathic inflammatory myopathies (IIMs) using Patient-Reported Outcome Measurement Information System (PROMIS) instrument data obtained from the second COVID-19 vaccination in autoimmune disease (COVAD-2) e-survey database [2].

In response to the first point, we concur that our study has several limitations inherent to e-surveys. Importantly, the diagnosis of autoimmune diseases including IIM subtypes and disease activity were patient reported and not verified objectively, although efforts were made to identify the group with correct classifications as part of an extensive data cleaning protocol. This was facilitated by eliminating responses wherein the diagnosis was not verified by a specialist (neurologist, dermatologist or rheumatologist), individuals with incomplete entries and those who chose multiple IIM subtypes. In fact, the demographics of IIM patients were consistent with previous epidemiological studies, with a mean age of 59 years and women accounting for 71.6% of patients. Current treatments reported by IIM patients also aligned with what we had expected; for instance, methotrexate (20.6%) and mycophenolate mofetil (17.0%) were the most commonly used immunomodulatory agents, followed by hydroxychloroquine (14.4%), intravenous or subcutaneous immunoglobulin (13.5%) and rituximab (9.9%). While we agree that there may not be absolute certainty that responses came from patients themselves, the COVAD-2 survey was designed specifically for IIMs and was shared extensively among myositis patient support groups, wherein most members either have IIMs themselves or are their caregivers [3]. These groups have policies to screen out individuals who may not have the condition. We excluded pregnant and breastfeeding women, as pregnancy and delivery significantly affect HRQoL [4]. Social media–driven research is a crucial frontier in the emerging landscape of digital health and assumes an even larger role in the context of rare disease research [5].

In terms of the second point of assessing physical and mental health using an electronic questionnaire, HRQoL is a domain of patient-reported outcome measures (PROMs) that evaluates the effect of a disease on the physical and mental health of individuals living with chronic diseases, including IIMs [6]. By definition, HRQoL must be evaluated by the patients themselves, and it is crucial to note that objective disease activity measures such as those proposed by the International Myositis Assessment and Clinical Studies Group fall short of assessing patients’ lived experiences. PROMIS is a National Institutes of Health–funded initiative to develop and validate PROMs. PROMIS short forms are shown to have excellent feasibility and validity not only in daily clinical practice, but also in e-surveys [7]. A recent multicentre study in the USA, Myositis Patient Centered Tele-Research (My PACER), reported that the PROMIS physical function 20-item short form (PROMIS PF-20) demonstrated consistent psychometric properties, whether administered in-person or through smartphone- and web-based technology, highlighting the excellent feasibility of PROMIS short forms in telemedicine [8]. Given the study’s self-reported nature, our results require validation through prospective cohort studies with physician and patient entries, which would be the focus of the next phase of the COVAD studies.

The third point is regarding data handling and the appropriateness of controls. From a statistical point of view, the large sample size and comparator group may potentially adjust for inadvertent data entry errors. To address missing data, we conducted sensitivity analyses on variables with >10% missing values, excluding them from covariates, where results remained consistent. Finsterer [1] noted that many controls, defined as those without autoimmune diseases, had comorbidities and suggested excluding these controls from the analysis. Our large sample size allowed us to include all relevant comorbidities in the multivariable model, adjusting for their effects on HRQoL. Excluding controls with comorbidities would significantly reduce the sample size and remove older individuals, limiting the results’ generalizability. Importantly, patients with IIMs were more likely to have comorbidities compared with other disease groups or controls. Furthermore, several comorbidities were identified as independent factors associated with impaired physical and mental health. We believe multivariable analysis was the best way to assess the modification effect of comorbidities on HRQoL.

We thank Finsterer [1] for initiating the discussion and concur that e-surveys have limitations despite the unique advantage of a large global sample of IIM patients that allowed the assessment of HRQoL in understudied subtypes like immune-mediated necrotizing myopathies and overlap myositis. This breadth is rarely achieved in academic cohorts. Our findings underscore the importance of patients’ daily lived experiences and embody patient voices often overlooked in clinical practice.

Supplementary Material

rkae098_Supplementary_Data

Acknowledgments

The authors are grateful to all respondents for completing the questionnaire. The authors also thank the Myositis Association, Myositis India, Myositis UK, Myositis Support and Understanding, the Myositis Global Network, Deutsche Gesellschaft für Muskelkranke e. V. (DGM), Dutch and Swedish Myositis patient support groups, Cure JM, Cure IBM, Sjögren’s India Foundation, Patients Engage, Scleroderma India, Lupus UK, Lupus Sweden, Emirates Arthritis Foundation, EULAR PARE, ArLAR research group, AAAA patient group, Myositis Association of Australia, APLAR myositis special interest group, Thai Rheumatism Association, PANLAR, AFLAR NRAS, Anti-Synthetase Syndrome support group and various other patient support groups and organizations for their contribution to the dissemination of this survey. Finally, the authors wish to thank all members of the COVAD study group for their invaluable role in the data collection. No part of this manuscript has been copied or published elsewhere either in whole or in part.

Supplementary material

Supplementary material is available at Rheumatology Advances in Practice online.

Data availability

The datasets generated and/or analysed during the current study are not publicly available but are available from the corresponding author upon reasonable request.

Authors’ contributions

A.Y., M.K., V.A. and L.G. were responsible for conceptualization. A.Y. and L.G. were responsible for the investigation. L.G., V.A. and A.Y. were responsible for the methodology. L.G. was responsible for the software. V.A. and L.G. were responsible for validation. R.A., V.A. and L.G. were responsible for visualization. A.Y., M.K. and L.G. wrote the original draft. All authors were responsible for data curation and reviewed and edited the manuscript.

Funding

No specific funding was received from any funding body in the public, commercial and not-for profit sectors for this article.

Disclosure statement: M.K. has received consulting fees, speaking fees and research grants from AbbVie, Argenx, Asahi Kasei, AstraZeneca, Astellas, Boehringer Ingelheim, Chugai, Corbus, Eisai, GSK, Horizon, Janssen, Kissei, MBL, Mitsubishi Tanabe, Mochida, Nippon Shinyaku, Ono Pharmaceuticals, Pfizer and Taisho. The remaining authors have declared no conflicts of interest.
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References

1 Finsterer J. Comment on: Impaired health-related quality of life in idiopathic inflammatory myopathies: a cross-sectional analysis from the COVAD-2 e-survey. Rheumatol Adv Pract 2024;rkae097. 10.1093/rap/rkae097.
2 Yoshida A , LiY, MaroufyV et al Impaired health-related quality of life in idiopathic inflammatory myopathies: a cross-sectional analysis from the COVAD-2 e-survey. Rheumatol Adv Pract 2024;8 :rkae028.38524696
3 Fazal ZZ , SenP, JoshiM et al COVAD survey 2 long-term outcomes: unmet need and protocol. Rheumatol Int 2022;42 :2151–8.35964271
4 Andreoli L , SenP, LiniD et al COVID-19 vaccine safety during the antenatal period in women with idiopathic inflammatory myopathies. Rheumatology (Oxford) 2023;62 :e175–9.36370070
5 Kapoor KK , TamilmaniK, RanaNP et al Advances in social media research: past, present and future. Inf Syst Front 2018;20 :531–58.
6 DiRenzo D , BinghamCO3rd, MecoliCA. Patient-reported outcomes in adult idiopathic inflammatory myopathies. Curr Rheumatol Rep 2019;21 :62.31741079
7 Yoshida A , KimM, KuwanaM et al Impaired physical function in patients with idiopathic inflammatory myopathies: results from the multicentre COVAD patient-reported e-survey. Rheumatology (Oxford) 2023;62 :1204–15.35920795
8 Keret S , SilvaRL, ChandraT et al Patient reported outcome for physical function in idiopathic inflammatory myopathy. Rheumatology (Oxford) 2024;keae091.
