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What matters most to pediatric rheumatologists in deciding whether to discontinue biologics in a child with juvenile idiopathic arthritis? A best-worst scaling survey

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Abstract

Objectives

Care for JIA patients has been transformed in the biologics era; however, biologics carry important (although rare) risks and are costly. Flares after biological withdrawal are seen frequently, yet there is little clinical guidance to identify which patients in clinical remission can safely have their biologic discontinued (by stopping or tapering). We examined what characteristics of the child or their context are important to pediatric rheumatologists when making the decision to discuss withdrawal of biologics.

Methods

We conducted a survey including a best-worst scaling (BWS) exercise in pediatric rheumatologists who are part of the UCAN CAN-DU network to assess the relative importance of 14 previously identified characteristics. A balanced incomplete block design was used to generate choice tasks. Respondents evaluated 14 choice sets of 5 characteristics of a child with JIA and identified for each set which was the most and least important in the decision to offer withdrawal. Results were analyzed using conditional logit regression.

Results

Fifty-one (out of 79) pediatric rheumatologists participated (response rate 65%). The three most important characteristics were how challenging it was to achieve remission, history of established joint damage, and time spent in remission. The three least important characteristics were history of temporomandibular joint involvement, accessibility of biologics, and the patient’s age.

Conclusions

These findings give quantitative insight about factors important to pediatric rheumatologists’ decision-making about biologic withdrawal. In addition to high quality clinical evidence, further research is needed to understand the perspective of patients and families to inform shared decision-making about biologic withdrawal for JIA patients with clinically inactive disease.

Key Points

● What is already known on this topic—there is limited clinical guidance for pediatric rheumatologists in making decisions about biologic withdrawal for patients with juvenile idiopathic arthritis who are in clinical remission.

● What this study adds—this study quantitatively examined what characteristic of the child in clinical remission, or of their context, are most important to pediatric rheumatologists in deciding whether to offer withdrawal of biologics.

● How this study might affect research, practice or policy—understanding of these characteristics can provide useful information to other pediatric rheumatologists in making their decisions, and may guide areas to focus on for future research.

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Data availability

The ethics approval for this survey did not include sharing the data beyond the study team; therefore, we are unable to make this data available.

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Acknowledgements

This study was undertaken as part of the UCAN CAN-DU and UCAN CURE consortia. SB is supported by the Husky Energy Chair in Child and Maternal Health and the Alberta Children’s Hospital Foundation Chair in Pediatric Research. RSMY is supported by the Hak-Ming and Deborah Chiu Chair in Paediatric Translational Research. DAM is supported by the Arthur J.E. Child Chair in Rheumatology (2012–2022) and a Canada Research Chair in Health Systems and Services Research (2008–2018).

Funding

This work was supported by the Canadian Institutes for Health Research (Canada) (grant number 381280); Genome Canada (Canada) (grant number OGI-150); Genome Alberta (Canada), Ontario Genomics (Canada); The Arthritis Society (Canada) (grant number GC-18-001); the Hospital for Sick Children (Canada); the University of Calgary (Canada); ZonMw (the Netherlands) (grant number 848006001); and the Reumafonds (the Netherlands).

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Authors and Affiliations

  1. Department of Paediatrics, Cumming School of Medicine, University of Calgary, Calgary, Alberta, Canada

    Gillian R. Currie

  2. Department of Community Health Sciences, Cumming School of Medicine, University of Calgary, Calgary, Alberta, Canada

    Gillian R. Currie & Deborah A Marshall

  3. Health Research Innovation Centre, Room 3C56, 3280 Hospital Drive NW, Calgary, AB, T2N 4Z6, Canada

    Gillian R. Currie

  4. Department of Health Technology and Services Research, Faculty of Behavioural, Management and Social Sciences, Technical Medical Centre, University of Twente, Enschede, the Netherlands

    Catherina G.M. Groothuis-Oudshoorn, Michelle M. A. Kip & Maarten J. IJzerman

  5. Section of Rheumatology, Department of Paediatrics, Cumming School of Medicine, University of Calgary, Calgary, Alberta, Canada

    Marinka Twilt & Susanne M. Benseler

  6. Alberta Children’s Hospital Research Institute, University of Calgary, Calgary, Alberta, Canada

    Marinka Twilt, Susanne M. Benseler & Deborah A Marshall

  7. Department of Pediatric Immunology and Rheumatology, Wilhelmina Children’s Hospital/UMC Utrecht, Utrecht, the Netherlands

    Joost F. Swart, Sebastiaan J. Vastert & Nico M. Wulffraat

  8. Faculty of Medicine, Utrecht University, Utrecht, the Netherlands

    Joost F. Swart, Sebastiaan J. Vastert & Nico M. Wulffraat

  9. Departments of Paediatrics, Immunology and Medical Science, The Hospital for Sick Children, University of Toronto, Toronto, Canada

    Rae Yeung

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  1. Gillian R. Currie
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  2. Catherina G.M. Groothuis-Oudshoorn
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  11. Deborah A Marshall
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Contributions

GC and DM were involved in the conception and design of the study. CG-O and GC conducted the data analysis. All authors contributed to data interpretation. GC drafted the manuscript, and all others critically reviewed it. All authors read and approved the final manuscript.

Corresponding author

Correspondence to Gillian R. Currie.

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Competing interests

DM reports grants from Canadian Institutes of Health Research, Alberta innovates, and the Arthritis Society supports for attending meetings from Illumina and ISPOR, and consulting fees from Analytica. All are outside of the submitted work.

RY reports consulting fees from Novartis and Lily, outside the submitted work.

SV reports grants and personal fees from SOBI and Novartis during the conduct of the study, outside the submitted work.

JS reports consulting fees from Amgen, outside the submitted work.

NW reports honoraria from UBC for a lecture on vaccinations.

All other authors declare that they gave no competing interests.

Patient involvement

There was no direct patient involvement in this component of the research.

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Currie, G.R., Groothuis-Oudshoorn, C., Twilt, M. et al. What matters most to pediatric rheumatologists in deciding whether to discontinue biologics in a child with juvenile idiopathic arthritis? A best-worst scaling survey. Clin Rheumatol 42, 2173–2180 (2023). https://doi.org/10.1007/s10067-023-06616-6

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  • DOI: https://doi.org/10.1007/s10067-023-06616-6

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