Ideal serum non-ceruloplasmin bound copper prediction for long-term treated patients with Wilson disease: a nomogram model
PurposeThis study aimed to explore the factors associated with the optimal serum non-ceruloplasmin bound copper (NCBC) level and develop a flexible predictive model to guide lifelong therapy in Wilson disease (WD) and delay disease progression.MethodsWe retrospectively collected clinical data from 1...
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| Format: | Article |
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Frontiers Media S.A.
2023-11-01
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| Series: | Frontiers in Medicine |
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| Online Access: | https://www.frontiersin.org/articles/10.3389/fmed.2023.1275242/full |
| _version_ | 1797639634068439040 |
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| author | Zhuang Tao Pingping Yang Jiafeng Zhou Rui Wang Zhenzhen Jiang Hui Han Meixia Wang |
| author_facet | Zhuang Tao Pingping Yang Jiafeng Zhou Rui Wang Zhenzhen Jiang Hui Han Meixia Wang |
| author_sort | Zhuang Tao |
| collection | DOAJ |
| description | PurposeThis study aimed to explore the factors associated with the optimal serum non-ceruloplasmin bound copper (NCBC) level and develop a flexible predictive model to guide lifelong therapy in Wilson disease (WD) and delay disease progression.MethodsWe retrospectively collected clinical data from 144 patients hospitalized in the Encephalopathy Center of the first affiliated hospital of Anhui University of Chinese Medicine between May 2012 and April 2023. Independent variables were selected using variate COX and LASSO regressions, followed by multivariate COX regression analysis. A predictive nomogram was constructed and validated using the concordance index (C-index), calibration curves, and clinical decision curve analysis, of which nomogram pictures were utilized for model visualization.ResultsA total of 61 (42.36%) patients were included, with an average treatment duration of 55.0 (range, 28.0, 97.0) months. Multivariate regression analysis identified several independent risk factors for serum NCBC level, including age of diagnosis, clinical classification, laminin liver stiffness measurement, and copper to zinc ratio in 24-h urinary excretion. The C-index indicated moderate discriminative ability (48 months: 0.829, 60 months: 0.811, and 72 months: 0.819). The calibration curves showed good consistency and calibration; clinical decision curve analysis demonstrated clinically beneficial threshold probabilities at different time intervals.ConclusionThe predictive nomogram model can predict serum NCBC level; consequently, we recommend its use in clinical practice to delay disease progression and improve the clinical prognosis of WD. |
| first_indexed | 2024-03-11T13:20:23Z |
| format | Article |
| id | doaj.art-3c67b058c2e04d42a3f43a22563fc1af |
| institution | Directory Open Access Journal |
| issn | 2296-858X |
| language | English |
| last_indexed | 2024-03-11T13:20:23Z |
| publishDate | 2023-11-01 |
| publisher | Frontiers Media S.A. |
| record_format | Article |
| series | Frontiers in Medicine |
| spelling | doaj.art-3c67b058c2e04d42a3f43a22563fc1af2023-11-03T10:34:44ZengFrontiers Media S.A.Frontiers in Medicine2296-858X2023-11-011010.3389/fmed.2023.12752421275242Ideal serum non-ceruloplasmin bound copper prediction for long-term treated patients with Wilson disease: a nomogram modelZhuang Tao0Pingping Yang1Jiafeng Zhou2Rui Wang3Zhenzhen Jiang4Hui Han5Meixia Wang6Encephalopathy Center, The First Affiliated Hospital of Anhui University of Chinese Medicine, Hefei, Anhui, ChinaGraduate School, Anhui University of Chinese Medicine, Hefei, Anhui, ChinaGraduate School, Anhui University of Chinese Medicine, Hefei, Anhui, ChinaGraduate School, Anhui University of Chinese Medicine, Hefei, Anhui, ChinaGraduate School, Anhui University of Chinese Medicine, Hefei, Anhui, ChinaGraduate School, Anhui University of Chinese Medicine, Hefei, Anhui, ChinaEncephalopathy Center, The First Affiliated Hospital of Anhui University of Chinese Medicine, Hefei, Anhui, ChinaPurposeThis study aimed to explore the factors associated with the optimal serum non-ceruloplasmin bound copper (NCBC) level and develop a flexible predictive model to guide lifelong therapy in Wilson disease (WD) and delay disease progression.MethodsWe retrospectively collected clinical data from 144 patients hospitalized in the Encephalopathy Center of the first affiliated hospital of Anhui University of Chinese Medicine between May 2012 and April 2023. Independent variables were selected using variate COX and LASSO regressions, followed by multivariate COX regression analysis. A predictive nomogram was constructed and validated using the concordance index (C-index), calibration curves, and clinical decision curve analysis, of which nomogram pictures were utilized for model visualization.ResultsA total of 61 (42.36%) patients were included, with an average treatment duration of 55.0 (range, 28.0, 97.0) months. Multivariate regression analysis identified several independent risk factors for serum NCBC level, including age of diagnosis, clinical classification, laminin liver stiffness measurement, and copper to zinc ratio in 24-h urinary excretion. The C-index indicated moderate discriminative ability (48 months: 0.829, 60 months: 0.811, and 72 months: 0.819). The calibration curves showed good consistency and calibration; clinical decision curve analysis demonstrated clinically beneficial threshold probabilities at different time intervals.ConclusionThe predictive nomogram model can predict serum NCBC level; consequently, we recommend its use in clinical practice to delay disease progression and improve the clinical prognosis of WD.https://www.frontiersin.org/articles/10.3389/fmed.2023.1275242/fullWilson diseaseserum NCBCclinical predictive modelretrospective studyconcordance index |
| spellingShingle | Zhuang Tao Pingping Yang Jiafeng Zhou Rui Wang Zhenzhen Jiang Hui Han Meixia Wang Ideal serum non-ceruloplasmin bound copper prediction for long-term treated patients with Wilson disease: a nomogram model Frontiers in Medicine Wilson disease serum NCBC clinical predictive model retrospective study concordance index |
| title | Ideal serum non-ceruloplasmin bound copper prediction for long-term treated patients with Wilson disease: a nomogram model |
| title_full | Ideal serum non-ceruloplasmin bound copper prediction for long-term treated patients with Wilson disease: a nomogram model |
| title_fullStr | Ideal serum non-ceruloplasmin bound copper prediction for long-term treated patients with Wilson disease: a nomogram model |
| title_full_unstemmed | Ideal serum non-ceruloplasmin bound copper prediction for long-term treated patients with Wilson disease: a nomogram model |
| title_short | Ideal serum non-ceruloplasmin bound copper prediction for long-term treated patients with Wilson disease: a nomogram model |
| title_sort | ideal serum non ceruloplasmin bound copper prediction for long term treated patients with wilson disease a nomogram model |
| topic | Wilson disease serum NCBC clinical predictive model retrospective study concordance index |
| url | https://www.frontiersin.org/articles/10.3389/fmed.2023.1275242/full |
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