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Journal of Medical Case Reports
Published in: Journal of Medical Case Reports 1/2024

Open Access 01-12-2024 | Osteitis | Case report

Dramatic response of synovitis, acne, pustulosis, hyperostosis, and osteitis syndrome to tofacitinib monotherapy: a case report

Authors: Stéphanie Dierckx, Jean-François Nisolle, Yves Boutsen

Published in: Journal of Medical Case Reports | Issue 1/2024

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Abstract

Introduction

The synovitis, acne, pustulosis, hyperostosis, and osteitis (SAPHO) syndrome is a rare condition. Its treatment remains a challenge for clinicians, and often yields mixed results.

Case

We report the case of a 51-year-old Caucasian woman who presented with SAPHO syndrome with mainly axial involvement. She had been treated with sulfasalazine and anti-inflammatory drugs for many years without any success. A few weeks after starting treatment with tofacitinib, both clinical and biological parameters dramatically improved. Imaging also showed considerable regression of the vertebral and pelvic lesions. However, tofacitinib had to be discontinued due to the occurrence of pulmonary embolism. Consequently, recurrence of bone pain and biologic inflammation was rapidly observed.

Conclusions

Anti-JAKs are an interesting treatment option in the management of SAPHO syndrome that need further clinical trials and assessment for validating response.
Literature
1.
Yang Q, Zhao Y, Li C, Luo Y, Hao W, Zhang W. Case report: successful treatment of refractory SAPHO syndrome with the JAK inhibitor tofacitinib. Medicine (Baltimore). 2018;97: e11149.CrossRefPubMed
2.
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(4527):34.
3.
Cianci F, Zoli A, Gremese E, Ferraccioli G. Clinical heterogeneity of SAPHO syndrome: challenging diagnose and treatment. Clin Rheumatol. 2017;36(2151):8.
4.
Nguyen MT, Borchers A, Selmi C, Naguwa SM, Cheema G, Gershwin ME. The SAPHO syndrome. Semin Arthritis Rheum. 2012;42(254):65.
5.
Kahn MF, Bouvier M, Palazzo E, Tebib JG, Colson F. Sternoclavicular pustulotic osteitis (SAPHO). 20-year interval between skin and bone lesions. J Rheumatol. 1991;18(1104):8.
6.
Firinu D, Garcia-Larsen V, Manconi PE, Del Giacco SR. SAPHO syndrome: current developments and approaches to clinical treatment. Curr Rheumatol Rep. 2016;18:35.CrossRefPubMed
7.
Xu W, Li C, Zhao X, et al. Whole-spine computed tomography findings in SAPHO syndrome. J Rheumatol. 2017;44(648):54.
8.
Zhang LH, Han SB, Song L, Gao S, Zhao Q, Deng XL, Yuan HS. Comparative analysis and differentiation between SAPHO syndrome and spondyloarthropathies using whole-spine MRI. Clin Radiol. 2021;76(5):394.e9-394.e14.CrossRefPubMed
9.
Ben Abdelghani K, Dran DG, Gottenberg JE, Morel J, Sibilia J, Combe B. Tumor necrosis factor-alpha blockers in SAPHO syndrome. J Rheumatol. 2010;37(1699):1704.
10.
Wendling D, Aubin F, Verhoeven F, Prati C. IL-23/Th17 targeted therapies in SAPHO syndrome. A case series. Joint Bone Spine. 2017;84(733):5.
11.
Li C, Zhao Y, Zuo Y, et al. Efficacy of bisphosphonates in patients with synovitis, acne, pustulosis, hyperostosis, and osteitis syndrome: a prospective open study. Clin Exp Rheumatol. 2019;37(663):9.
12.
Guignard S, Job-Deslandre C, Sayag-Boukris V, Kahan A. Pamidronate treatment in SAPHO syndrome. Joint Bone Spine. 2002;69(392):6.
13.
Li Y, Huo J, Cao Y, Yu M, Zhang Y, Li Z, Li C, Zhang W. Efficacy of tofacitinib in synovitis, acne, pustulosis, hyperostosis and osteitis syndrome: a pilot study with clinical and MRI evaluation. Ann Rheum Dis. 2020;79(9):1255–7.CrossRefPubMed
14.
Yuan F, Luo J, Yang Q. SAPHO syndrome complicated by ankylosing spondylitis successfully treated with tofacitinib: a case report. Front Immunol. 2022;25(13): 911922.CrossRef
15.
Xie W, Huang H, Zhang Z. Off-label use of tofacitinib: a potential treatment option for SAPHO syndrome. Ann Rheum Dis. 2022;81: e91.CrossRefPubMed
Metadata
Title
Dramatic response of synovitis, acne, pustulosis, hyperostosis, and osteitis syndrome to tofacitinib monotherapy: a case report
Authors
Stéphanie Dierckx
Jean-François Nisolle
Yves Boutsen
Publication date
01-12-2024
Publisher
BioMed Central
Published in
Journal of Medical Case Reports / Issue 1/2024
Electronic ISSN: 1752-1947
DOI
https://doi.org/10.1186/s13256-024-04366-w

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