. 2023 Jul 13;21(1):70.

doi: 10.1186/s12969-023-00839-2.

The successes and challenges of harmonising juvenile idiopathic arthritis (JIA) datasets to create a large-scale JIA data resource

Saskia Lawson-Tovey^{1

2}, Samantha Louise Smith³, Nophar Geifman⁴, Stephanie Shoop-Worrall^{5

6}, Sandra Ng⁷, Michael R Barnes⁷, Lucy R Wedderburn^{8

9}, Kimme L Hyrich^{10

5}; CLUSTER consortium

Collaborators, Affiliations

Collaborators

CLUSTER consortium:
Melissa Kartawinata, Zoe Wanstall, Bethany R Jebson, Alyssia McNeece, Elizabeth Ralph, Vasiliki Alexiou, Fatjon Dekaj, Aline Kimonyo, Fatema Merali, Emma Sumner, Emily Robinson, Freya L Feilding, Andrew Dick, Michael W Beresford, Emil Carlsson, Joanna Fairlie, Jenna F Gritzfeld, Athimalaipet Ramanan, Teresa Duerr, Stephen Eyre, Soumya Raychaudhuri, Andrew Morris, Annie Yarwood, Samantha Smith, John Bowes, Paul Martin, Melissa Tordoff, Michael Stadler, Wendy Thomson, Damian Tarasek, Chris Wallace, Wei-Yu Lin, Sarah Clarke, Toby Kent, Thierry Sornasse, Daniela Dastros-Pitei, Sumanta Mukherjee, Jacqui Roberts, Rami Kallala, Helen Neale, John Ioannou, Hussein Al-Mossawi

Affiliations

¹ Centre for Genetics and Genomics Versus Arthritis, The University of Manchester, Manchester, UK. saskia.lawson-tovey@manchester.ac.uk.
² National Institute of Health Research Manchester Biomedical Research Centre, Manchester University NHS Foundation Trust, Manchester Academic Health Science Centre, Manchester, UK. saskia.lawson-tovey@manchester.ac.uk.
³ Centre for Genetics and Genomics Versus Arthritis, The University of Manchester, Manchester, UK.
⁴ School of Health Sciences, Faculty of Health and Medical Sciences, The University of Surrey, Guildford, UK.
⁵ Centre for Epidemiology Versus Arthritis, The University of Manchester, Manchester, UK.
⁶ Centre for Health Informatics, The University of Manchester, Manchester, UK.
⁷ Centre for Translational Bioinformatics, William Harvey Research Institute, Bart's and the London School of Medicine and Dentistry, Queen Mary University London, London, UK.
⁸ Centre for Adolescent Rheumatology Versus Arthritis at UCL, University College London Hospitals and Great Ormond Street Hospital, London, UK.
⁹ Infection, Inflammation and Rheumatology, UCL Great Ormond Street Institute of Child Health, London, UK.
¹⁰ National Institute of Health Research Manchester Biomedical Research Centre, Manchester University NHS Foundation Trust, Manchester Academic Health Science Centre, Manchester, UK.

PMID: 37438749
PMCID: PMC10339614
DOI: 10.1186/s12969-023-00839-2

The successes and challenges of harmonising juvenile idiopathic arthritis (JIA) datasets to create a large-scale JIA data resource

Saskia Lawson-Tovey et al. Pediatr Rheumatol Online J. 2023.

. 2023 Jul 13;21(1):70.

doi: 10.1186/s12969-023-00839-2.

Authors

Collaborators

CLUSTER consortium:
Melissa Kartawinata, Zoe Wanstall, Bethany R Jebson, Alyssia McNeece, Elizabeth Ralph, Vasiliki Alexiou, Fatjon Dekaj, Aline Kimonyo, Fatema Merali, Emma Sumner, Emily Robinson, Freya L Feilding, Andrew Dick, Michael W Beresford, Emil Carlsson, Joanna Fairlie, Jenna F Gritzfeld, Athimalaipet Ramanan, Teresa Duerr, Stephen Eyre, Soumya Raychaudhuri, Andrew Morris, Annie Yarwood, Samantha Smith, John Bowes, Paul Martin, Melissa Tordoff, Michael Stadler, Wendy Thomson, Damian Tarasek, Chris Wallace, Wei-Yu Lin, Sarah Clarke, Toby Kent, Thierry Sornasse, Daniela Dastros-Pitei, Sumanta Mukherjee, Jacqui Roberts, Rami Kallala, Helen Neale, John Ioannou, Hussein Al-Mossawi

Affiliations

¹ Centre for Genetics and Genomics Versus Arthritis, The University of Manchester, Manchester, UK. saskia.lawson-tovey@manchester.ac.uk.
² National Institute of Health Research Manchester Biomedical Research Centre, Manchester University NHS Foundation Trust, Manchester Academic Health Science Centre, Manchester, UK. saskia.lawson-tovey@manchester.ac.uk.
³ Centre for Genetics and Genomics Versus Arthritis, The University of Manchester, Manchester, UK.
⁴ School of Health Sciences, Faculty of Health and Medical Sciences, The University of Surrey, Guildford, UK.
⁵ Centre for Epidemiology Versus Arthritis, The University of Manchester, Manchester, UK.
⁶ Centre for Health Informatics, The University of Manchester, Manchester, UK.
⁷ Centre for Translational Bioinformatics, William Harvey Research Institute, Bart's and the London School of Medicine and Dentistry, Queen Mary University London, London, UK.
⁸ Centre for Adolescent Rheumatology Versus Arthritis at UCL, University College London Hospitals and Great Ormond Street Hospital, London, UK.
⁹ Infection, Inflammation and Rheumatology, UCL Great Ormond Street Institute of Child Health, London, UK.
¹⁰ National Institute of Health Research Manchester Biomedical Research Centre, Manchester University NHS Foundation Trust, Manchester Academic Health Science Centre, Manchester, UK.

PMID: 37438749
PMCID: PMC10339614
DOI: 10.1186/s12969-023-00839-2

Abstract

Background: CLUSTER is a UK consortium focussed on precision medicine research in JIA/JIA-Uveitis. As part of this programme, a large-scale JIA data resource was created by harmonizing and pooling existing real-world studies. Here we present challenges and progress towards creation of this unique large JIA dataset.

Methods: Four real-world studies contributed data; two clinical datasets of JIA patients starting first-line methotrexate (MTX) or tumour necrosis factor inhibitors (TNFi) were created. Variables were selected based on a previously developed core dataset, and encrypted NHS numbers were used to identify children contributing similar data across multiple studies.

Results: Of 7013 records (from 5435 individuals), 2882 (1304 individuals) represented the same child across studies. The final datasets contain 2899 (MTX) and 2401 (TNFi) unique patients; 1018 are in both datasets. Missingness ranged from 10 to 60% and was not improved through harmonisation.

Conclusions: Combining data across studies has achieved dataset sizes rarely seen in JIA, invaluable to progressing research. Losing variable specificity and missingness, and their impact on future analyses requires further consideration.

Keywords: Children and young people; Data harmonisation; JIA.

PubMed Disclaimer

Conflict of interest statement

SLT, SS, NG, SSW, SN have nothing to disclose. MRB reports grant income from Abbvie unrelated to this work. LRW reports non-personal consulting fees from Pfizer unrelated to this work; LRW is supported by the NIHR Great Ormond Street Biomedical Research Centre. KLH reports she has received non-personal speaker’s fees from Abbvie and grant income from BMS and Pfizer, all unrelated to this manuscript; KLH is supported by the NIHR Manchester Biomedical Research Centre.

Figures

**Fig. 1**
Distribution of individuals with more than one record at any timepoint (“duplicates”) across CLUSTER studies

**Fig. 2**
Percentage of missingness across key variables in the CLUSTER MTX and TNF datasets *MTX* Methotrexate, *TNF* Tumour necrosis factor inhibitors, T1 Timepoint 1 (closest values to baseline; allowed -3 months to drug start), T2 Timepoint 2 (closest value to 6 months after drug start; allowed 3–12 months after drug start), *JIA* Juvenile idiopathic arthritis, RF Rheumatoid factor, *HLA B27* Human leukocyte antigen B27, *ANA* Anti-nuclear antibody, *CHAQ* Childhood health assessment questionnaire, *ESR* Erythrocyte sedimentation rate, *CRP* C-reactive protein, *VAS* Visual analogue scale

See this image and copyright information in PMC

References

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1. Doiron D, Burton P, Marcon Y, Gaye A, Wolffenbuttel BHR, Perola M, et al. Data harmonization and federated analysis of population-based studies: The BioSHaRE project. Emerg Themes Epidemiol. 2013 Nov 21 [cited 2021 Mar 12];10:12. Available from: https://ete-online.biomedcentral.com/articles/10.1186/1742-7622-10-12 - PMC - PubMed
1. CLUSTER Consortium. [cited 2021 Mar 4]. Available from: https://www.clusterconsortium.org.uk/
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The successes and challenges of harmonising juvenile idiopathic arthritis (JIA) datasets to create a large-scale JIA data resource

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The successes and challenges of harmonising juvenile idiopathic arthritis (JIA) datasets to create a large-scale JIA data resource

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Conflict of interest statement

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