Development and validation of the AF score for diagnosis of adult-onset Still's disease in fever of unknown origin

Objective: To develop and validate a diagnostic score to identify adult-onset Still's disease (AOSD) in fever of unknown origin (FUO). Methods: A single center, retrospective case-control study of inpatients with FUO from January 2018 to December 2021. Using clinical and laboratory data from 17...

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Main Authors: Shuni Ying, Duo Lv, Dingxian Zhu, Sheng Li, Yuwei Ding, Chuanyin Sun, Yu Shi, Hong Fang, Jianjun Qiao
Format: Article
Language:English
Published: Elsevier 2023-01-01
Series:Journal of Translational Autoimmunity
Subjects:
Online Access:http://www.sciencedirect.com/science/article/pii/S2589909022000454
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author Shuni Ying
Duo Lv
Dingxian Zhu
Sheng Li
Yuwei Ding
Chuanyin Sun
Yu Shi
Hong Fang
Jianjun Qiao
author_facet Shuni Ying
Duo Lv
Dingxian Zhu
Sheng Li
Yuwei Ding
Chuanyin Sun
Yu Shi
Hong Fang
Jianjun Qiao
author_sort Shuni Ying
collection DOAJ
description Objective: To develop and validate a diagnostic score to identify adult-onset Still's disease (AOSD) in fever of unknown origin (FUO). Methods: A single center, retrospective case-control study of inpatients with FUO from January 2018 to December 2021. Using clinical and laboratory data from 178 cases with AOSD and 486 cases with FUO, we developed an AOSD/FUO (AF) score with a Bayesian Model Averaging approach. AF score and Yamaguchi's criteria were evaluated by sensitivity, specificity, accuracy, and positive/negative predictive value for diagnosis of AOSD in developmental and validation samples. Results: Persistent pruritic eruptions (PPEs) in patients with rashes was higher in AOSD group than FUO group (52.3% vs 7.4%; P < 0.01). PPEs yielded a specificity of 97.5% and a sensitivity of 44.9%. AF score = PPEs × 3.795+Evanescent rash × 2.774+Serum ferritin × 1.678+Myalgia × 0.958+Neutrophil count × 0.185+Platelet count × 0.004. A cut-off value ≥ 5.245 revealed the maximizing sensitivity of 88.7% and specificity of 95.8% in discriminating AOSD from FUO in the validation group. And AF score improved the accuracy from 82.6% to 93.3% compared with Yamaguchi's criteria. Conclusions: We developed and validated a new score which can identify AOSD in FUO with higher classification accuracy than Yamaguchi's criteria. Future multi-centric prospective studies need to be designed to confirm the diagnosis value of AF score.
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spelling doaj.art-d1e67e4124f4415d832a0aab974353172023-06-19T04:29:15ZengElsevierJournal of Translational Autoimmunity2589-90902023-01-016100184Development and validation of the AF score for diagnosis of adult-onset Still's disease in fever of unknown originShuni Ying0Duo Lv1Dingxian Zhu2Sheng Li3Yuwei Ding4Chuanyin Sun5Yu Shi6Hong Fang7Jianjun Qiao8Department of Dermatology, The First Affiliated Hospital, Zhejiang University School of Medicine, Hangzhou, ChinaDepartment of Clinical Pharmacy, The First Affiliated Hospital, Zhejiang University School of Medicine, Hangzhou, ChinaDepartment of Dermatology, The First Affiliated Hospital, Zhejiang University School of Medicine, Hangzhou, ChinaDepartment of Dermatology, The First Affiliated Hospital, Zhejiang University School of Medicine, Hangzhou, ChinaDepartment of Dermatology, The First Affiliated Hospital, Zhejiang University School of Medicine, Hangzhou, China; Department of Dermatology, Shaoxing People's Hospital (Shaoxing Hospital, Zhejiang University School of Medicine), Shaoxing, ChinaDepartment of Rheumatology, The First Affiliated Hospital, Zhejiang University School of Medicine, Hangzhou, ChinaDepartment of Infectious Diseases, The First Affiliated Hospital, Zhejiang University School of Medicine, Hangzhou, China; Corresponding author.Department of Dermatology, The First Affiliated Hospital, Zhejiang University School of Medicine, Hangzhou, China; Corresponding author.Department of Dermatology, The First Affiliated Hospital, Zhejiang University School of Medicine, Hangzhou, China; Corresponding author.Objective: To develop and validate a diagnostic score to identify adult-onset Still's disease (AOSD) in fever of unknown origin (FUO). Methods: A single center, retrospective case-control study of inpatients with FUO from January 2018 to December 2021. Using clinical and laboratory data from 178 cases with AOSD and 486 cases with FUO, we developed an AOSD/FUO (AF) score with a Bayesian Model Averaging approach. AF score and Yamaguchi's criteria were evaluated by sensitivity, specificity, accuracy, and positive/negative predictive value for diagnosis of AOSD in developmental and validation samples. Results: Persistent pruritic eruptions (PPEs) in patients with rashes was higher in AOSD group than FUO group (52.3% vs 7.4%; P < 0.01). PPEs yielded a specificity of 97.5% and a sensitivity of 44.9%. AF score = PPEs × 3.795+Evanescent rash × 2.774+Serum ferritin × 1.678+Myalgia × 0.958+Neutrophil count × 0.185+Platelet count × 0.004. A cut-off value ≥ 5.245 revealed the maximizing sensitivity of 88.7% and specificity of 95.8% in discriminating AOSD from FUO in the validation group. And AF score improved the accuracy from 82.6% to 93.3% compared with Yamaguchi's criteria. Conclusions: We developed and validated a new score which can identify AOSD in FUO with higher classification accuracy than Yamaguchi's criteria. Future multi-centric prospective studies need to be designed to confirm the diagnosis value of AF score.http://www.sciencedirect.com/science/article/pii/S2589909022000454Adult-onset Still's diseaseFever of unknown originPersistent pruritic eruptionsClassification criteriaBayesian method
spellingShingle Shuni Ying
Duo Lv
Dingxian Zhu
Sheng Li
Yuwei Ding
Chuanyin Sun
Yu Shi
Hong Fang
Jianjun Qiao
Development and validation of the AF score for diagnosis of adult-onset Still's disease in fever of unknown origin
Journal of Translational Autoimmunity
Adult-onset Still's disease
Fever of unknown origin
Persistent pruritic eruptions
Classification criteria
Bayesian method
title Development and validation of the AF score for diagnosis of adult-onset Still's disease in fever of unknown origin
title_full Development and validation of the AF score for diagnosis of adult-onset Still's disease in fever of unknown origin
title_fullStr Development and validation of the AF score for diagnosis of adult-onset Still's disease in fever of unknown origin
title_full_unstemmed Development and validation of the AF score for diagnosis of adult-onset Still's disease in fever of unknown origin
title_short Development and validation of the AF score for diagnosis of adult-onset Still's disease in fever of unknown origin
title_sort development and validation of the af score for diagnosis of adult onset still s disease in fever of unknown origin
topic Adult-onset Still's disease
Fever of unknown origin
Persistent pruritic eruptions
Classification criteria
Bayesian method
url http://www.sciencedirect.com/science/article/pii/S2589909022000454
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