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Case report: successful treatment of refractory SAPHO syndrome with the JAK inhibitor tofacitinib

INTRODUCTION: Synovitis, acne, pustulosis, hyperostosis, and osteitis (SAPHO) syndrome is an autoinflammatory disorder without standardized treatment. Janus kinase (JAK) inhibitors can block a range of cytokines and might possess significant anti-inflammatory activity. Here, we report the first case...

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Autores principales: Yang, Qiao, Zhao, Yumo, Li, Chen, Luo, Yaping, Hao, Weixin, Zhang, Wen
Formato: Online Artículo Texto
Lenguaje:English
Publicado: Wolters Kluwer Health 2018
Materias:
Acceso en línea:https://www.ncbi.nlm.nih.gov/pmc/articles/PMC6024477/
https://www.ncbi.nlm.nih.gov/pubmed/29924019
http://dx.doi.org/10.1097/MD.0000000000011149
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author Yang, Qiao
Zhao, Yumo
Li, Chen
Luo, Yaping
Hao, Weixin
Zhang, Wen
author_facet Yang, Qiao
Zhao, Yumo
Li, Chen
Luo, Yaping
Hao, Weixin
Zhang, Wen
author_sort Yang, Qiao
collection PubMed
description INTRODUCTION: Synovitis, acne, pustulosis, hyperostosis, and osteitis (SAPHO) syndrome is an autoinflammatory disorder without standardized treatment. Janus kinase (JAK) inhibitors can block a range of cytokines and might possess significant anti-inflammatory activity. Here, we report the first case of efficacious treatment of refractory SAPHO syndrome with the JAK inhibitor tofacitinib. CASE PRESENTATION: A 44-year-old woman presented with arthralgia in the right wrist and complained of having difficulty in doing housework. Symptoms were unresponsiveness to nonsteroidal anti-inflammatory drugs, disease-modifying antirheumatic drugs, and tumor necrosis factor inhibitors. A diagnosis of SAPHO syndrome was made based on previous dermatological and osteoarticular manifestations and bone scintigraphy findings. Oral treatment with tofacitinib at 5 mg twice daily in combination with the basic methotrexate treatment was initiated. After 4 weeks of using tofacitinib, the patient reported marked improvement of symptoms and also reported being competent in completing housework. CONCLUSIONS: The efficacy of JAK inhibitors in treating refractory SAPHO syndrome should be noted.
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spelling pubmed-60244772018-07-03 Case report: successful treatment of refractory SAPHO syndrome with the JAK inhibitor tofacitinib Yang, Qiao Zhao, Yumo Li, Chen Luo, Yaping Hao, Weixin Zhang, Wen Medicine (Baltimore) Research Article INTRODUCTION: Synovitis, acne, pustulosis, hyperostosis, and osteitis (SAPHO) syndrome is an autoinflammatory disorder without standardized treatment. Janus kinase (JAK) inhibitors can block a range of cytokines and might possess significant anti-inflammatory activity. Here, we report the first case of efficacious treatment of refractory SAPHO syndrome with the JAK inhibitor tofacitinib. CASE PRESENTATION: A 44-year-old woman presented with arthralgia in the right wrist and complained of having difficulty in doing housework. Symptoms were unresponsiveness to nonsteroidal anti-inflammatory drugs, disease-modifying antirheumatic drugs, and tumor necrosis factor inhibitors. A diagnosis of SAPHO syndrome was made based on previous dermatological and osteoarticular manifestations and bone scintigraphy findings. Oral treatment with tofacitinib at 5 mg twice daily in combination with the basic methotrexate treatment was initiated. After 4 weeks of using tofacitinib, the patient reported marked improvement of symptoms and also reported being competent in completing housework. CONCLUSIONS: The efficacy of JAK inhibitors in treating refractory SAPHO syndrome should be noted. Wolters Kluwer Health 2018-06-22 /pmc/articles/PMC6024477/ /pubmed/29924019 http://dx.doi.org/10.1097/MD.0000000000011149 Text en Copyright © 2018 the Author(s). Published by Wolters Kluwer Health, Inc. http://creativecommons.org/licenses/by-nc/4.0 This is an open access article distributed under the terms of the Creative Commons Attribution-Non Commercial License 4.0 (CCBY-NC), where it is permissible to download, share, remix, transform, and buildup the work provided it is properly cited. The work cannot be used commercially without permission from the journal. http://creativecommons.org/licenses/by-nc/4.0
spellingShingle Research Article
Yang, Qiao
Zhao, Yumo
Li, Chen
Luo, Yaping
Hao, Weixin
Zhang, Wen
Case report: successful treatment of refractory SAPHO syndrome with the JAK inhibitor tofacitinib
title Case report: successful treatment of refractory SAPHO syndrome with the JAK inhibitor tofacitinib
title_full Case report: successful treatment of refractory SAPHO syndrome with the JAK inhibitor tofacitinib
title_fullStr Case report: successful treatment of refractory SAPHO syndrome with the JAK inhibitor tofacitinib
title_full_unstemmed Case report: successful treatment of refractory SAPHO syndrome with the JAK inhibitor tofacitinib
title_short Case report: successful treatment of refractory SAPHO syndrome with the JAK inhibitor tofacitinib
title_sort case report: successful treatment of refractory sapho syndrome with the jak inhibitor tofacitinib
topic Research Article
url https://www.ncbi.nlm.nih.gov/pmc/articles/PMC6024477/
https://www.ncbi.nlm.nih.gov/pubmed/29924019
http://dx.doi.org/10.1097/MD.0000000000011149
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