TY - JOUR
T1 - What matters most to pediatric rheumatologists in deciding whether to discontinue biologics in a child with juvenile idiopathic arthritis? A best-worst scaling survey
AU - Currie, Gillian R.
AU - Groothuis-Oudshoorn, Catherina G.M.
AU - Twilt, Marinka
AU - Kip, Michelle M.A.
AU - IJzerman, Maarten J.
AU - Benseler, Susanne M.
AU - Swart, Joost F.
AU - Vastert, Sebastiaan J.
AU - Wulffraat, Nico M.
AU - Yeung, Rae
AU - Marshall, Deborah A.
N1 - Funding Information:
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.
Funding Information:
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 Information:
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 Information:
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).
Publisher Copyright:
© 2023, The Author(s), under exclusive licence to International League of Associations for Rheumatology (ILAR).
PY - 2023/8
Y1 - 2023/8
N2 - 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.
AB - 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.
KW - Arthritis, Juvenile
KW - Biological therapy
KW - Economics
KW - 2023 OA procedure
UR - http://www.scopus.com/inward/record.url?scp=85159648937&partnerID=8YFLogxK
U2 - 10.1007/s10067-023-06616-6
DO - 10.1007/s10067-023-06616-6
M3 - Article
AN - SCOPUS:85159648937
SN - 0770-3198
VL - 42
SP - 2173
EP - 2180
JO - Clinical rheumatology
JF - Clinical rheumatology
IS - 8
ER -