Validation of prediction models of severe disease course and non-achievement of remission in juvenile idiopathic arthritis: part 1—results of the Canadian model in the Nordic cohort
Abstract Background Models to predict disease course and long-term outcome based on clinical characteristics at disease onset may guide early treatment strategies in juvenile idiopathic arthritis (JIA). Before a prediction model can be recommended for use in clinical practice, it needs to be validat...
| Main Authors: | , , , , , , , , , , , , , , |
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| Format: | Article |
| Language: | English |
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BMC
2019-12-01
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| Series: | Arthritis Research & Therapy |
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| Online Access: | https://doi.org/10.1186/s13075-019-2060-2 |
| _version_ | 1818432741049368576 |
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| author | Veronika Rypdal Jaime Guzman Andrew Henrey Thomas Loughin Mia Glerup Ellen Dalen Arnstad Kristiina Aalto Marite Rygg Susan Nielsen Troels Herlin Anders Fasth Lillemor Berntson Martin Rypdal Ellen Nordal for the ReACCh-Out and NoSPeR Investigators |
| author_facet | Veronika Rypdal Jaime Guzman Andrew Henrey Thomas Loughin Mia Glerup Ellen Dalen Arnstad Kristiina Aalto Marite Rygg Susan Nielsen Troels Herlin Anders Fasth Lillemor Berntson Martin Rypdal Ellen Nordal for the ReACCh-Out and NoSPeR Investigators |
| author_sort | Veronika Rypdal |
| collection | DOAJ |
| description | Abstract Background Models to predict disease course and long-term outcome based on clinical characteristics at disease onset may guide early treatment strategies in juvenile idiopathic arthritis (JIA). Before a prediction model can be recommended for use in clinical practice, it needs to be validated in a different cohort than the one used for building the model. The aim of the current study was to validate the predictive performance of the Canadian prediction model developed by Guzman et al. and the Nordic model derived from Rypdal et al. to predict severe disease course and non-achievement of remission in Nordic patients with JIA. Methods The Canadian and Nordic multivariable logistic regression models were evaluated in the Nordic JIA cohort for prediction of non-achievement of remission, and the data-driven outcome denoted severe disease course. A total of 440 patients in the Nordic cohort with a baseline visit and an 8-year visit were included. The Canadian prediction model was first externally validated exactly as published. Both the Nordic and Canadian models were subsequently evaluated with repeated fine-tuning of model coefficients in training sets and testing in disjoint validation sets. The predictive performances of the models were assessed with receiver operating characteristic curves and C-indices. A model with a C-index above 0.7 was considered useful for clinical prediction. Results The Canadian prediction model had excellent predictive ability and was comparable in performance to the Nordic model in predicting severe disease course in the Nordic JIA cohort. The Canadian model yielded a C-index of 0.85 (IQR 0.83–0.87) for prediction of severe disease course and a C-index of 0.66 (0.63–0.68) for prediction of non-achievement of remission when applied directly. The median C-indices after fine-tuning were 0.85 (0.80–0.89) and 0.69 (0.65–0.73), respectively. Internal validation of the Nordic model for prediction of severe disease course resulted in a median C-index of 0.90 (0.86–0.92). Conclusions External validation of the Canadian model and internal validation of the Nordic model with severe disease course as outcome confirm their predictive abilities. Our findings suggest that predicting long-term remission is more challenging than predicting severe disease course. |
| first_indexed | 2024-12-14T16:10:01Z |
| format | Article |
| id | doaj.art-34d130f399494ce1a9690f020a4c78a5 |
| institution | Directory Open Access Journal |
| issn | 1478-6362 |
| language | English |
| last_indexed | 2024-12-14T16:10:01Z |
| publishDate | 2019-12-01 |
| publisher | BMC |
| record_format | Article |
| series | Arthritis Research & Therapy |
| spelling | doaj.art-34d130f399494ce1a9690f020a4c78a52022-12-21T22:55:01ZengBMCArthritis Research & Therapy1478-63622019-12-0121111010.1186/s13075-019-2060-2Validation of prediction models of severe disease course and non-achievement of remission in juvenile idiopathic arthritis: part 1—results of the Canadian model in the Nordic cohortVeronika Rypdal0Jaime Guzman1Andrew Henrey2Thomas Loughin3Mia Glerup4Ellen Dalen Arnstad5Kristiina Aalto6Marite Rygg7Susan Nielsen8Troels Herlin9Anders Fasth10Lillemor Berntson11Martin Rypdal12Ellen Nordal13for the ReACCh-Out and NoSPeR InvestigatorsDepartment of Pediatrics, University Hospital of North NorwayDepartment of Pediatrics, BC Children’s Hospital and University of British ColumbiaDepartment of Statistics and Actuarial Sciences, Simon Fraser UniversityDepartment of Statistics and Actuarial Sciences, Simon Fraser UniversityDepartment of Pediatrics, Aarhus University HospitalDepartment of Clinical and Molecular Medicine, NTNU - Norwegian University of Science and TechnologyDepartment of Pediatrics, Helsinki University Hospital, University of HelsinkiDepartment of Clinical and Molecular Medicine, NTNU - Norwegian University of Science and TechnologyDepartment of Pediatrics, Rigshospitalet Copenhagen University HospitalDepartment of Pediatrics, Aarhus University HospitalDepartment of Pediatrics, Institute of Clinical Sciences, Sahlgrenska Academy, University of GothenburgDepartment of Women’s and Children’s Health, Uppsala UniversityDepartment of Mathematics and Statistics, UIT – The Arctic University of NorwayDepartment of Pediatrics, University Hospital of North NorwayAbstract Background Models to predict disease course and long-term outcome based on clinical characteristics at disease onset may guide early treatment strategies in juvenile idiopathic arthritis (JIA). Before a prediction model can be recommended for use in clinical practice, it needs to be validated in a different cohort than the one used for building the model. The aim of the current study was to validate the predictive performance of the Canadian prediction model developed by Guzman et al. and the Nordic model derived from Rypdal et al. to predict severe disease course and non-achievement of remission in Nordic patients with JIA. Methods The Canadian and Nordic multivariable logistic regression models were evaluated in the Nordic JIA cohort for prediction of non-achievement of remission, and the data-driven outcome denoted severe disease course. A total of 440 patients in the Nordic cohort with a baseline visit and an 8-year visit were included. The Canadian prediction model was first externally validated exactly as published. Both the Nordic and Canadian models were subsequently evaluated with repeated fine-tuning of model coefficients in training sets and testing in disjoint validation sets. The predictive performances of the models were assessed with receiver operating characteristic curves and C-indices. A model with a C-index above 0.7 was considered useful for clinical prediction. Results The Canadian prediction model had excellent predictive ability and was comparable in performance to the Nordic model in predicting severe disease course in the Nordic JIA cohort. The Canadian model yielded a C-index of 0.85 (IQR 0.83–0.87) for prediction of severe disease course and a C-index of 0.66 (0.63–0.68) for prediction of non-achievement of remission when applied directly. The median C-indices after fine-tuning were 0.85 (0.80–0.89) and 0.69 (0.65–0.73), respectively. Internal validation of the Nordic model for prediction of severe disease course resulted in a median C-index of 0.90 (0.86–0.92). Conclusions External validation of the Canadian model and internal validation of the Nordic model with severe disease course as outcome confirm their predictive abilities. Our findings suggest that predicting long-term remission is more challenging than predicting severe disease course.https://doi.org/10.1186/s13075-019-2060-2Juvenile idiopathic arthritisPredictionValidationOutcome researchRemission |
| spellingShingle | Veronika Rypdal Jaime Guzman Andrew Henrey Thomas Loughin Mia Glerup Ellen Dalen Arnstad Kristiina Aalto Marite Rygg Susan Nielsen Troels Herlin Anders Fasth Lillemor Berntson Martin Rypdal Ellen Nordal for the ReACCh-Out and NoSPeR Investigators Validation of prediction models of severe disease course and non-achievement of remission in juvenile idiopathic arthritis: part 1—results of the Canadian model in the Nordic cohort Arthritis Research & Therapy Juvenile idiopathic arthritis Prediction Validation Outcome research Remission |
| title | Validation of prediction models of severe disease course and non-achievement of remission in juvenile idiopathic arthritis: part 1—results of the Canadian model in the Nordic cohort |
| title_full | Validation of prediction models of severe disease course and non-achievement of remission in juvenile idiopathic arthritis: part 1—results of the Canadian model in the Nordic cohort |
| title_fullStr | Validation of prediction models of severe disease course and non-achievement of remission in juvenile idiopathic arthritis: part 1—results of the Canadian model in the Nordic cohort |
| title_full_unstemmed | Validation of prediction models of severe disease course and non-achievement of remission in juvenile idiopathic arthritis: part 1—results of the Canadian model in the Nordic cohort |
| title_short | Validation of prediction models of severe disease course and non-achievement of remission in juvenile idiopathic arthritis: part 1—results of the Canadian model in the Nordic cohort |
| title_sort | validation of prediction models of severe disease course and non achievement of remission in juvenile idiopathic arthritis part 1 results of the canadian model in the nordic cohort |
| topic | Juvenile idiopathic arthritis Prediction Validation Outcome research Remission |
| url | https://doi.org/10.1186/s13075-019-2060-2 |
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