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Does hand involvement in systemic sclerosis limit completion of patient-reported outcome measures?

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Abstract

The objective of this analysis is to examine whether the severity of systemic sclerosis (SSc)-hand involvement influences patient-reported outcome measure (PROM) completion rate in a US cohort of early disease. Participants included SSc patients with less than 5 years disease duration consented and enrolled in the Collaborative, National, Quality, and Efficacy Registry (CONQUER) between June 2018 and December 2019. Participants’ socio-demographics, hand clinical features (severe modified Rodnan skin score, presence of small joint contractures, acro-osteolysis, calcinosis, and digital ulcers), and completion rates of seven PROMs including a Resource Use Questionnaire were analyzed. Cohort characteristics and baseline PROM completion were evaluated. Multivariable logistic regression assessed the relationship between hand limitations and PROM incompletion at several time points using generalized estimating equations. At the time of data lock, 339 CONQUER subjects had a total of 600 visits available for analysis. Calcinosis (odds ratio [OR] 6.35, confidence interval [CI] 2.41–16.73 and acro-osteolysis OR 3.88 (1.57–9.55) were significantly associated with incomplete PROM. The Resource Use Questionnaire was the PROM most commonly not completed. Increasing age was correlated with resource use questionnaire incompletion rate. Acro-osteolysis and calcinosis were associated with lower PROM completion rates in a US SSc cohort, independent of the length of the questionnaires or the modality of administration (electronic or paper). Resource Use Questionnaires are important for understanding the economic impact and burden of chronic disease; however, in this study, it had lower completion rates than PROMs devoted to clinical variables.

Key points

•Multiple strategies are needed to ensure optimal completion of PROM in longitudinal cohort studies. Even if patients request electronic surveys, we have found it is important to follow up incomplete surveys with paper forms provided at the time of a clinical visit.

•The Resource Utilization Questionnaire was lengthy and prone to non-completion in the younger population.

•Acro-osteolysis and calcinosis were associated with reduced PROM completion rates.

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Funding

TF receives research funding from the National Institutes of Health (K23AR067889) and the U.S. Department of Veterans Affairs (I01 CX001183). LC receives research funding from the Scleroderma Research Foundation and Boehringer Ingelheim. DK was supported by NIH/NIAMS K24AR063120-07. LH receives funding from the Scleroderma Research Foundation. AS receives research funding from the Scleroderma Research Foundation and NIH/NIAMS R01 AR073208. SA receives grants from Scleroderma Research Foundation and NIH/NIAMS R01 AR073284. EJB receives research funding from the NIH/NIAMS (K23AR075112).

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Authors and Affiliations

  1. Department of Internal Medicine, Division of Rheumatology, University of Utah and Salt Lake Veterans Affair Medical Center, 1900 E 30 N, SOM 4b200, Salt Lake City, UT, 84132, USA

    Tracy M. Frech

  2. Department of Pediatrics, University of Utah, Salt Lake City, UT, USA

    John M. VanBuren & Emily Startup

  3. The University of Texas Health Science Center at Houston, Houston, TX, USA

    Shervin Assassi

  4. Department of Medicine, Vagelos College of Physicians and Surgeons, Columbia University Irving Medical Center, New York, NY, USA

    Elana J. Bernstein

  5. Harvard Medical School and Massachusetts General Hospital, Boston, MA, USA

    Flavia V. Castelino

  6. Department of Medicine and Dermatology, Division of Rheumatology, Stanford University and Palo Alto Veterans Affairs Health Care System, Stanford, CA, USA

    Lorinda Chung

  7. Department of Medicine, Division of Rheumatology, Northwestern University Feinberg School of Medicine, Chicago, IL, USA

    Chase Correia

  8. Hospital for Special Surgery, New York, NY, USA

    Jessica K. Gordon

  9. Division of Rheumatology and Immunology, Medical University of South Carolina, Charleston, SC, USA

    Faye N. Hant

  10. Department of Medicine, Division of Rheumatology, Johns Hopkins University School of Medicine, Baltimore, MD, USA

    Laura Hummers & Ami A. Shah

  11. University of Michigan Scleroderma Program, Ann Arbor, MI, USA

    Dinesh Khanna

  12. Division of Rheumatology, University of Pennsylvania, Philadelphia, PA, USA

    Nora Sandorfi

  13. Division of Rheumatology, The George Washington University School of Medicine and Health Sciences, Washington, DC, USA

    Victoria K. Shanmugam

  14. Georgetown University School of Medicine, Washington, DC, USA

    Virginia Steen

  15. Scleroderma Research Foundation, San Francisco, CA, USA

    Luke Evnin

Authors
  1. Tracy M. Frech
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  2. John M. VanBuren
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  3. Emily Startup
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  4. Shervin Assassi
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  5. Elana J. Bernstein
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  6. Flavia V. Castelino
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  7. Lorinda Chung
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  8. Chase Correia
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  9. Jessica K. Gordon
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  10. Faye N. Hant
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  11. Laura Hummers
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  12. Dinesh Khanna
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  13. Nora Sandorfi
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  14. Ami A. Shah
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  15. Victoria K. Shanmugam
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  16. Virginia Steen
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  17. Luke Evnin
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Corresponding author

Correspondence to Tracy M. Frech.

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Frech, T.M., VanBuren, J.M., Startup, E. et al. Does hand involvement in systemic sclerosis limit completion of patient-reported outcome measures?. Clin Rheumatol 40, 965–971 (2021). https://doi.org/10.1007/s10067-020-05467-9

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  • DOI: https://doi.org/10.1007/s10067-020-05467-9

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