Li B, Li GW, Xue L, Chen YY. Rapid remission of refractory synovitis, acne, pustulosis, hyperostosis, and osteitis syndrome in response to the Janus kinase inhibitor tofacitinib: A case report. World J Clin Cases 2020; 8(19): 4527-4534 [PMID: 33083414 DOI: 10.12998/wjcc.v8.i19.4527]
Corresponding Author of This Article
Luan Xue, MD, Chief Doctor, Professor, Department of Rheumatology, Yueyang Hospital of Integrated Traditional Chinese and Western Medicine, Shanghai University of Traditional Chinese Medicine, 15th floor, Building 1, No. 110 Ganhe Road, Hongkou District, Shanghai 200437, China. xelco@163.com
Research Domain of This Article
Rheumatology
Article-Type of This Article
Case Report
Open-Access Policy of This Article
This article is an open-access article which was selected by an in-house editor and fully peer-reviewed by external reviewers. It is distributed in accordance with the Creative Commons Attribution Non Commercial (CC BY-NC 4.0) license, which permits others to distribute, remix, adapt, build upon this work non-commercially, and license their derivative works on different terms, provided the original work is properly cited and the use is non-commercial. See: http://creativecommons.org/licenses/by-nc/4.0/
World J Clin Cases. Oct 6, 2020; 8(19): 4527-4534 Published online Oct 6, 2020. doi: 10.12998/wjcc.v8.i19.4527
Rapid remission of refractory synovitis, acne, pustulosis, hyperostosis, and osteitis syndrome in response to the Janus kinase inhibitor tofacitinib: A case report
Ben Li, Guan-Wu Li, Luan Xue, Yue-Ying Chen
Ben Li, Luan Xue, Yue-Ying Chen, Department of Rheumatology, Yueyang Hospital of Integrated Traditional Chinese and Western Medicine, Shanghai University of Traditional Chinese Medicine, Shanghai 200437, China
Guan-Wu Li, Department of Radiology, Yueyang Hospital of Integrated Traditional Chinese and Western Medicine, Shanghai University of Traditional Chinese Medicine, Shanghai 200437, China
Author contributions: Li B collected and analyzed the clinical data and literature, and drafted, designed, wrote and revised the manuscript; Xue L collected the clinical data during treatment, and designed and revised the manuscript; Li GW performed the radiologic diagnosis, imaging material evaluation, and revision of the manuscript; Chen YY collected the clinical data of previous treatment in another hospital and drafted the manuscript; All authors approved the manuscript and approved submission.
Informed consent statement: Informed written consent was obtained from the patient for publication of this report and any accompanying images.
Conflict-of-interest statement: The authors declare that they have no conflict of interest.
CARE Checklist (2016) statement: The authors have read the CARE Checklist (2016), and the manuscript was prepared and revised according to the CARE Checklist (2016).
Open-Access: This article is an open-access article that was selected by an in-house editor and fully peer-reviewed by external reviewers. It is distributed in accordance with the Creative Commons Attribution NonCommercial (CC BY-NC 4.0) license, which permits others to distribute, remix, adapt, build upon this work non-commercially, and license their derivative works on different terms, provided the original work is properly cited and the use is non-commercial. See: http://creativecommons.org/licenses/by-nc/4.0/
Corresponding author: Luan Xue, MD, Chief Doctor, Professor, Department of Rheumatology, Yueyang Hospital of Integrated Traditional Chinese and Western Medicine, Shanghai University of Traditional Chinese Medicine, 15th floor, Building 1, No. 110 Ganhe Road, Hongkou District, Shanghai 200437, China. xelco@163.com
Received: May 14, 2020 Peer-review started: May 14, 2020 First decision: June 7, 2020 Revised: June 18, 2020 Accepted: August 29, 2020 Article in press: August 29, 2020 Published online: October 6, 2020 Processing time: 136 Days and 15.3 Hours
Abstract
BACKGROUND
Synovitis, acne, pustulosis, hyperostosis, and osteitis (SAPHO) syndrome is a rare autoinflammatory disease for which clinical treatment has not been standardized. Janus kinase (JAK) inhibitors represent a novel therapeutic option for rheumatoid arthritis, psoriatic arthritis, and some other autoinflammatory diseases. However, the clinical utility of JAK inhibitors in treating SAPHO syndrome has not been thoroughly investigated. In this study, we describe a patient with SAPHO syndrome who failed to respond to conventional treatment but demonstrated a remarkable and rapid response to the JAK inhibitor tofacitinib.
CASE SUMMARY
A 62-year-old female patient presented with swelling and pain at the sternoclavicular joints, back pain that limited her activities, arthralgia in the right knee, and cutaneous lesions. Her symptoms were unresponsive to nonsteroidal anti-inflammatory drugs, disease-modifying antirheumatic drugs, Tripterygium wilfordii hook f, and bisphosphonates. SAPHO syndrome was diagnosed in accordance with dermatological and osteoarticular manifestations and abnormal inflammatory factors. Multiple image studies have illustrated bone lesions and pathological fractures of vertebral bodies. Oral treatment with tofacitinib at 5 mg twice daily with methotrexate and bisphosphonates was initiated. The patient reported that her pain symptoms were relieved after 3 d and her cutaneous lesions were reduced after 4 wk of treatment. Vertebral lesions were improved after 6 mo on tofacitinib. No serious adverse effects were noted.
CONCLUSION
JAK inhibitor therapy may be a promising strategy to treat SAPHO syndrome.
Core Tip: Synovitis, acne, pustulosis, hyperostosis, and osteitis (SAPHO) syndrome is a rare autoinflammatory disease for which clinical treatment has not been standardized. We describe a patient with SAPHO syndrome who failed to respond to conventional treatment but demonstrated a remarkable and rapid response to the Janus kinase inhibitor tofacitinib. This case highlights that Janus kinase inhibitor therapy may be a promising strategy to treat SAPHO syndrome.
