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

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. We report the case of a 51-year-old Caucasian woman who presented with SAPHO syndrome with mainly axial involvement. She...

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Bibliographic Details
Published in:Journal of medical case reports 2024-02, Vol.18 (1), p.57-57, Article 57
Main Authors: Dierckx, Stéphanie, Nisolle, Jean-François, Boutsen, Yves
Format: Article
Language:English
Subjects:
Acne
Anti-inflammatory agents
Arthritis
Bisphosphonates
Blood tests
Case report
Case reports
Disodium pamidronate
Exostosis
JAK-inhibitors
Kinases
Magnetic resonance imaging
Nonsteroidal anti-inflammatory drugs
Patients
Pelvis
Pulmonary embolism
Reimbursement
Rheumatic diseases
Sacroiliitis
SAPHO syndrome
Skin
Sulfasalazine
Synovitis
TNF inhibitors
Tofacitinib
Tomography
Tumor necrosis factor-TNF
Vertebrae
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Summary: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. 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. Anti-JAKs are an interesting treatment option in the management of SAPHO syndrome that need further clinical trials and assessment for validating response.
ISSN:1752-1947
1752-1947
DOI:10.1186/s13256-024-04366-w