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Failure of tocilizumab in treating two patients with refractory SAPHO syndrome: a case report

Synovitis, acne, pustulosis, hyperostosis, and osteitis (SAPHO) syndrome is a rare autoinflammatory disease with no standard treatment. Interleukin (IL)-6 inhibitors represent a novel therapeutic option for rheumatoid arthritis and some autoinflammatory diseases. However, the clinical utility of IL-...

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Autores principales: Sun, Xiao-Chuan, Liu, Shuang, Li, Chen, Zhang, Shuo, Wang, Mu, Shi, Xiao-Hua, Hao, Wei-Xin, Zhang, Wen
Formato: Online Artículo Texto
Lenguaje:English
Publicado: SAGE Publications 2018
Materias:
Acceso en línea:https://www.ncbi.nlm.nih.gov/pmc/articles/PMC6300969/
https://www.ncbi.nlm.nih.gov/pubmed/30428761
http://dx.doi.org/10.1177/0300060518806105
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author Sun, Xiao-Chuan
Liu, Shuang
Li, Chen
Zhang, Shuo
Wang, Mu
Shi, Xiao-Hua
Hao, Wei-Xin
Zhang, Wen
author_facet Sun, Xiao-Chuan
Liu, Shuang
Li, Chen
Zhang, Shuo
Wang, Mu
Shi, Xiao-Hua
Hao, Wei-Xin
Zhang, Wen
author_sort Sun, Xiao-Chuan
collection PubMed
description Synovitis, acne, pustulosis, hyperostosis, and osteitis (SAPHO) syndrome is a rare autoinflammatory disease with no standard treatment. Interleukin (IL)-6 inhibitors represent a novel therapeutic option for rheumatoid arthritis and some autoinflammatory diseases. However, the clinical utility of IL-6 inhibitors in treating SAPHO syndrome has been poorly investigated. In the present report, we describe two patients with SAPHO syndrome that was unresponsive to conventional treatment. Tocilizumab, an anti-IL-6 receptor monoclonal antibody, was putatively administered according to positive IL-6 immunohistochemical staining in biopsied bone tissues. However, the disease continued to progress, and new-onset or worsening skin lesions were noted with transient neutropenia. These cases demonstrate that tocilizumab may not be an ideal option for treating SAPHO syndrome.
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spelling pubmed-63009692019-01-08 Failure of tocilizumab in treating two patients with refractory SAPHO syndrome: a case report Sun, Xiao-Chuan Liu, Shuang Li, Chen Zhang, Shuo Wang, Mu Shi, Xiao-Hua Hao, Wei-Xin Zhang, Wen J Int Med Res Case Reports Synovitis, acne, pustulosis, hyperostosis, and osteitis (SAPHO) syndrome is a rare autoinflammatory disease with no standard treatment. Interleukin (IL)-6 inhibitors represent a novel therapeutic option for rheumatoid arthritis and some autoinflammatory diseases. However, the clinical utility of IL-6 inhibitors in treating SAPHO syndrome has been poorly investigated. In the present report, we describe two patients with SAPHO syndrome that was unresponsive to conventional treatment. Tocilizumab, an anti-IL-6 receptor monoclonal antibody, was putatively administered according to positive IL-6 immunohistochemical staining in biopsied bone tissues. However, the disease continued to progress, and new-onset or worsening skin lesions were noted with transient neutropenia. These cases demonstrate that tocilizumab may not be an ideal option for treating SAPHO syndrome. SAGE Publications 2018-11-14 2018-12 /pmc/articles/PMC6300969/ /pubmed/30428761 http://dx.doi.org/10.1177/0300060518806105 Text en © The Author(s) 2018 http://creativecommons.org/licenses/by-nc/4.0/ Creative Commons Non Commercial CC BY-NC: This article is distributed under the terms of the Creative Commons Attribution-NonCommercial 4.0 License (http://www.creativecommons.org/licenses/by-nc/4.0/) which permits non-commercial use, reproduction and distribution of the work without further permission provided the original work is attributed as specified on the SAGE and Open Access pages (https://us.sagepub.com/en-us/nam/open-access-at-sage).
spellingShingle Case Reports
Sun, Xiao-Chuan
Liu, Shuang
Li, Chen
Zhang, Shuo
Wang, Mu
Shi, Xiao-Hua
Hao, Wei-Xin
Zhang, Wen
Failure of tocilizumab in treating two patients with refractory SAPHO syndrome: a case report
title Failure of tocilizumab in treating two patients with refractory SAPHO syndrome: a case report
title_full Failure of tocilizumab in treating two patients with refractory SAPHO syndrome: a case report
title_fullStr Failure of tocilizumab in treating two patients with refractory SAPHO syndrome: a case report
title_full_unstemmed Failure of tocilizumab in treating two patients with refractory SAPHO syndrome: a case report
title_short Failure of tocilizumab in treating two patients with refractory SAPHO syndrome: a case report
title_sort failure of tocilizumab in treating two patients with refractory sapho syndrome: a case report
topic Case Reports
url https://www.ncbi.nlm.nih.gov/pmc/articles/PMC6300969/
https://www.ncbi.nlm.nih.gov/pubmed/30428761
http://dx.doi.org/10.1177/0300060518806105
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