Development of CARRA/PReS-endorsed consensus Core and Expanded Datasets in childhood-onset systemic lupus erythematosus for international registry-based research

Rebecca E. Sadun; Jennifer C. Cooper; Alexandre Belot; Tadej Avcin; Amita Aggarwal; Jenny Ainsworth; Alisha Akinsete; Stacy P. Ardoin; Michael W. Beresford; Lynette Bortey; Hermine I. Brunner; Joyce C. Chang; Coziana Ciurtin; Ashley Daftary; Barbara Eberhard; Candace H. Feldman; Christian M. Hedrich; Aimee O. Hersh; Linda T. Hiraki; David A. Isenberg; Sylvia Kamphuis; Andrea M. Knight; Lou Lambert; Deborah M. Levy; Stephen D. Marks; Naomi Maxwell; Angela Migowa; Katharine Moore; Seza Ozen; Rosalind Ramsey-Goldman; Angelo Ravelli; Bryce B. Reeve; Tamar B. Rubinstein; Claudia Saad-Magalhaes; Sujata Sawhney; Laura E. Schanberg; Emily Von Scheven; Christiaan Scott; Mary Beth Son; Gladys Tony; Elissa R. Weitzman; Scott E. Wenderfer; Alisha Woodside; Laura B. Lewandowski; Eve M.D. Smith

doi:10.1136/ard-2024-226528

Development of CARRA/PReS-endorsed consensus Core and Expanded Datasets in childhood-onset systemic lupus erythematosus for international registry-based research

Rebecca E. Sadun, Jennifer C. Cooper, Alexandre Belot, Tadej Avcin, Amita Aggarwal, Jenny Ainsworth, Alisha Akinsete, Stacy P. Ardoin, Michael W. Beresford, Lynette Bortey, Hermine I. Brunner, Joyce C. Chang, Coziana Ciurtin, Ashley Daftary, Barbara Eberhard, Candace H. Feldman, Christian M. Hedrich, Aimee O. Hersh, Linda T. Hiraki, David A. IsenbergSylvia Kamphuis, Andrea M. Knight, Lou Lambert, Deborah M. Levy, Stephen D. Marks, Naomi Maxwell, Angela Migowa, Katharine Moore, Seza Ozen, Rosalind Ramsey-Goldman, Angelo Ravelli, Bryce B. Reeve, Tamar B. Rubinstein, Claudia Saad-Magalhaes, Sujata Sawhney, Laura E. Schanberg, Emily Von Scheven, Christiaan Scott, Mary Beth Son, Gladys Tony, Elissa R. Weitzman, Scott E. Wenderfer, Alisha Woodside, Laura B. Lewandowski, Eve M.D. Smith

Medicine, Rheumatology Division

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

Abstract

Objectives: Childhood-onset systemic lupus erythematosus (cSLE), representing 15%-20% of individuals with SLE, has been difficult to study globally due to differences between registries. This initiative, supported by Childhood Arthritis Rheumatology Research Alliance (CARRA) and Paediatric Rheumatology European Society (PReS), aims to create Core and Expanded cSLE Datasets to standardise and enhance research worldwide. Methods: 21 international cSLE experts and 4 patients participated in a Delphi process (questionnaires, 2 topic-specific focus groups and 3 virtual consensus meetings) to create 2 standardised cSLE datasets. The Core cSLE Dataset was designed to include data essential to meaningful clinical research across many settings. The Expanded cSLE Dataset was designed for centres able to consistently collect data to address broader research questions. Final data items for the Core and Expanded datasets were determined by consensus defined as >80% agreement) using an adapted nominal group technique and voting. Results: The resulting Core cSLE Dataset contains 46 items, including demographics, clinical features, laboratory results, medications and significant adverse events. The Expanded cSLE Dataset adds 26 additional items and includes patient-reported outcomes. Consensus was also achieved regarding the frequency and time points for data collection: baseline, quarterly follow-up visits, annually and flare visits. Conclusion: Standardised Core and Expanded cSLE Datasets for registry-based international cSLE research were defined through the consensus of global experts and patient/caregiver representatives, endorsed by CARRA and PReS. These datasets incorporate disease-specific and patient-specific features, optimised for diverse settings to facilitate international collaborative research for children and adolescents with SLE worldwide.

Original language	English (US)
Journal	Annals of the rheumatic diseases
DOIs	https://doi.org/10.1136/ard-2024-226528
State	Accepted/In press - 2024

Funding

This project was funded by the CARRA/PReS Collaborative Award, and the authors wish to acknowledge the ongoing Arthritis Foundation financial support of CARRA. The authors would like to thank Katherine Cowan, Anne Dennos, Fran\u00E7ois Hofer, Carla Roberts and Laura Whitty for their invaluable contributions. In addition, we are grateful to CARRA leadership and the CARRA SLE Committee, PReS leadership and SLE working party for their support of this project. We would like to thank the CARRA Registry, the JIR Lupus Cohort (Pedialup) study, and the UK JSLE Cohort Study and Repository, led by and the UK\u2019s Experimental Arthritis Treatment Centre for Children, for providing access to their data dictionaries and experience in developing and collecting real-world data in cSLE. Furthermore, we are grateful to all those individuals and centres who support these studies, and to all the patients and families who have helped inform and contribute data by participating in them. This research was funded by the inaugural CARRA/PReS Collaborative Research Award (JXR30630). LBL was funded by the NIAMS Intramural Program.

Keywords

Child
Lupus Erythematosus, Systemic
Lupus Nephritis
Outcome Assessment, Health Care
Patient Reported Outcome Measures

ASJC Scopus subject areas

Rheumatology
Immunology and Allergy
Immunology
General Biochemistry, Genetics and Molecular Biology

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10.1136/ard-2024-226528

Cite this

Sadun, R. E., Cooper, J. C., Belot, A., Avcin, T., Aggarwal, A., Ainsworth, J., Akinsete, A., Ardoin, S. P., Beresford, M. W., Bortey, L., Brunner, H. I., Chang, J. C., Ciurtin, C., Daftary, A., Eberhard, B., Feldman, C. H., Hedrich, C. M., Hersh, A. O., Hiraki, L. T., ... Smith, E. M. D. (Accepted/In press). Development of CARRA/PReS-endorsed consensus Core and Expanded Datasets in childhood-onset systemic lupus erythematosus for international registry-based research. Annals of the rheumatic diseases. https://doi.org/10.1136/ard-2024-226528

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title = "Development of CARRA/PReS-endorsed consensus Core and Expanded Datasets in childhood-onset systemic lupus erythematosus for international registry-based research",

abstract = "Objectives: Childhood-onset systemic lupus erythematosus (cSLE), representing 15%-20% of individuals with SLE, has been difficult to study globally due to differences between registries. This initiative, supported by Childhood Arthritis Rheumatology Research Alliance (CARRA) and Paediatric Rheumatology European Society (PReS), aims to create Core and Expanded cSLE Datasets to standardise and enhance research worldwide. Methods: 21 international cSLE experts and 4 patients participated in a Delphi process (questionnaires, 2 topic-specific focus groups and 3 virtual consensus meetings) to create 2 standardised cSLE datasets. The Core cSLE Dataset was designed to include data essential to meaningful clinical research across many settings. The Expanded cSLE Dataset was designed for centres able to consistently collect data to address broader research questions. Final data items for the Core and Expanded datasets were determined by consensus defined as >80% agreement) using an adapted nominal group technique and voting. Results: The resulting Core cSLE Dataset contains 46 items, including demographics, clinical features, laboratory results, medications and significant adverse events. The Expanded cSLE Dataset adds 26 additional items and includes patient-reported outcomes. Consensus was also achieved regarding the frequency and time points for data collection: baseline, quarterly follow-up visits, annually and flare visits. Conclusion: Standardised Core and Expanded cSLE Datasets for registry-based international cSLE research were defined through the consensus of global experts and patient/caregiver representatives, endorsed by CARRA and PReS. These datasets incorporate disease-specific and patient-specific features, optimised for diverse settings to facilitate international collaborative research for children and adolescents with SLE worldwide.",

keywords = "Child, Lupus Erythematosus, Systemic, Lupus Nephritis, Outcome Assessment, Health Care, Patient Reported Outcome Measures",

author = "Sadun, {Rebecca E.} and Cooper, {Jennifer C.} and Alexandre Belot and Tadej Avcin and Amita Aggarwal and Jenny Ainsworth and Alisha Akinsete and Ardoin, {Stacy P.} and Beresford, {Michael W.} and Lynette Bortey and Brunner, {Hermine I.} and Chang, {Joyce C.} and Coziana Ciurtin and Ashley Daftary and Barbara Eberhard and Feldman, {Candace H.} and Hedrich, {Christian M.} and Hersh, {Aimee O.} and Hiraki, {Linda T.} and Isenberg, {David A.} and Sylvia Kamphuis and Knight, {Andrea M.} and Lou Lambert and Levy, {Deborah M.} and Marks, {Stephen D.} and Naomi Maxwell and Angela Migowa and Katharine Moore and Seza Ozen and Rosalind Ramsey-Goldman and Angelo Ravelli and Reeve, {Bryce B.} and Rubinstein, {Tamar B.} and Claudia Saad-Magalhaes and Sujata Sawhney and Schanberg, {Laura E.} and {Von Scheven}, Emily and Christiaan Scott and Son, {Mary Beth} and Gladys Tony and Weitzman, {Elissa R.} and Wenderfer, {Scott E.} and Alisha Woodside and Lewandowski, {Laura B.} and Smith, {Eve M.D.}",

note = "Publisher Copyright: {\textcopyright} Author(s) (or their employer(s)) 2024.",

year = "2024",

doi = "10.1136/ard-2024-226528",

language = "English (US)",

journal = "Annals of the rheumatic diseases",

issn = "0003-4967",

publisher = "BMJ Publishing Group",

}

Sadun, RE, Cooper, JC, Belot, A, Avcin, T, Aggarwal, A, Ainsworth, J, Akinsete, A, Ardoin, SP, Beresford, MW, Bortey, L, Brunner, HI, Chang, JC, Ciurtin, C, Daftary, A, Eberhard, B, Feldman, CH, Hedrich, CM, Hersh, AO, Hiraki, LT, Isenberg, DA, Kamphuis, S, Knight, AM, Lambert, L, Levy, DM, Marks, SD, Maxwell, N, Migowa, A, Moore, K, Ozen, S, Ramsey-Goldman, R, Ravelli, A, Reeve, BB, Rubinstein, TB, Saad-Magalhaes, C, Sawhney, S, Schanberg, LE, Von Scheven, E, Scott, C, Son, MB, Tony, G, Weitzman, ER, Wenderfer, SE, Woodside, A, Lewandowski, LB & Smith, EMD 2024, 'Development of CARRA/PReS-endorsed consensus Core and Expanded Datasets in childhood-onset systemic lupus erythematosus for international registry-based research', Annals of the rheumatic diseases. https://doi.org/10.1136/ard-2024-226528

TY - JOUR

T1 - Development of CARRA/PReS-endorsed consensus Core and Expanded Datasets in childhood-onset systemic lupus erythematosus for international registry-based research

AU - Sadun, Rebecca E.

AU - Cooper, Jennifer C.

AU - Belot, Alexandre

AU - Avcin, Tadej

AU - Aggarwal, Amita

AU - Ainsworth, Jenny

AU - Akinsete, Alisha

AU - Ardoin, Stacy P.

AU - Beresford, Michael W.

AU - Bortey, Lynette

AU - Brunner, Hermine I.

AU - Chang, Joyce C.

AU - Ciurtin, Coziana

AU - Daftary, Ashley

AU - Eberhard, Barbara

AU - Feldman, Candace H.

AU - Hedrich, Christian M.

AU - Hersh, Aimee O.

AU - Hiraki, Linda T.

AU - Isenberg, David A.

AU - Kamphuis, Sylvia

AU - Knight, Andrea M.

AU - Lambert, Lou

AU - Levy, Deborah M.

AU - Marks, Stephen D.

AU - Maxwell, Naomi

AU - Migowa, Angela

AU - Moore, Katharine

AU - Ozen, Seza

AU - Ramsey-Goldman, Rosalind

AU - Ravelli, Angelo

AU - Reeve, Bryce B.

AU - Rubinstein, Tamar B.

AU - Saad-Magalhaes, Claudia

AU - Sawhney, Sujata

AU - Schanberg, Laura E.

AU - Von Scheven, Emily

AU - Scott, Christiaan

AU - Son, Mary Beth

AU - Tony, Gladys

AU - Weitzman, Elissa R.

AU - Wenderfer, Scott E.

AU - Woodside, Alisha

AU - Lewandowski, Laura B.

AU - Smith, Eve M.D.

PY - 2024

Y1 - 2024

N2 - Objectives: Childhood-onset systemic lupus erythematosus (cSLE), representing 15%-20% of individuals with SLE, has been difficult to study globally due to differences between registries. This initiative, supported by Childhood Arthritis Rheumatology Research Alliance (CARRA) and Paediatric Rheumatology European Society (PReS), aims to create Core and Expanded cSLE Datasets to standardise and enhance research worldwide. Methods: 21 international cSLE experts and 4 patients participated in a Delphi process (questionnaires, 2 topic-specific focus groups and 3 virtual consensus meetings) to create 2 standardised cSLE datasets. The Core cSLE Dataset was designed to include data essential to meaningful clinical research across many settings. The Expanded cSLE Dataset was designed for centres able to consistently collect data to address broader research questions. Final data items for the Core and Expanded datasets were determined by consensus defined as >80% agreement) using an adapted nominal group technique and voting. Results: The resulting Core cSLE Dataset contains 46 items, including demographics, clinical features, laboratory results, medications and significant adverse events. The Expanded cSLE Dataset adds 26 additional items and includes patient-reported outcomes. Consensus was also achieved regarding the frequency and time points for data collection: baseline, quarterly follow-up visits, annually and flare visits. Conclusion: Standardised Core and Expanded cSLE Datasets for registry-based international cSLE research were defined through the consensus of global experts and patient/caregiver representatives, endorsed by CARRA and PReS. These datasets incorporate disease-specific and patient-specific features, optimised for diverse settings to facilitate international collaborative research for children and adolescents with SLE worldwide.

AB - Objectives: Childhood-onset systemic lupus erythematosus (cSLE), representing 15%-20% of individuals with SLE, has been difficult to study globally due to differences between registries. This initiative, supported by Childhood Arthritis Rheumatology Research Alliance (CARRA) and Paediatric Rheumatology European Society (PReS), aims to create Core and Expanded cSLE Datasets to standardise and enhance research worldwide. Methods: 21 international cSLE experts and 4 patients participated in a Delphi process (questionnaires, 2 topic-specific focus groups and 3 virtual consensus meetings) to create 2 standardised cSLE datasets. The Core cSLE Dataset was designed to include data essential to meaningful clinical research across many settings. The Expanded cSLE Dataset was designed for centres able to consistently collect data to address broader research questions. Final data items for the Core and Expanded datasets were determined by consensus defined as >80% agreement) using an adapted nominal group technique and voting. Results: The resulting Core cSLE Dataset contains 46 items, including demographics, clinical features, laboratory results, medications and significant adverse events. The Expanded cSLE Dataset adds 26 additional items and includes patient-reported outcomes. Consensus was also achieved regarding the frequency and time points for data collection: baseline, quarterly follow-up visits, annually and flare visits. Conclusion: Standardised Core and Expanded cSLE Datasets for registry-based international cSLE research were defined through the consensus of global experts and patient/caregiver representatives, endorsed by CARRA and PReS. These datasets incorporate disease-specific and patient-specific features, optimised for diverse settings to facilitate international collaborative research for children and adolescents with SLE worldwide.

KW - Child

KW - Lupus Erythematosus, Systemic

KW - Lupus Nephritis

KW - Outcome Assessment, Health Care

KW - Patient Reported Outcome Measures

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UR - http://www.scopus.com/inward/citedby.url?scp=85206611689&partnerID=8YFLogxK

U2 - 10.1136/ard-2024-226528

DO - 10.1136/ard-2024-226528

M3 - Article

C2 - 39379141

AN - SCOPUS:85206611689

SN - 0003-4967

JO - Annals of the rheumatic diseases

JF - Annals of the rheumatic diseases

ER -

Development of CARRA/PReS-endorsed consensus Core and Expanded Datasets in childhood-onset systemic lupus erythematosus for international registry-based research

Abstract

Funding

Keywords

ASJC Scopus subject areas

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