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作者: 范薇
单位: 厦门医学院附属第二医院

摘要


This case aims to explore an optimized treatment strategy for SAPHO syndrome (Synovitis, Acne, Pustulosis, Hyperostosis, and Osteitis).Due to its rarity, most treatment strategies are derived from case reports or retrospective studies, and no standardized treatment currently exists. We report a 40-year-old male patient admitted with neck, chest, and lumbar back pain lasting two years, accompanied by pustular rashes and itching on both hands and feet, ultimately diagnosed with SAPHO syndrome improved after treatment, to provide clinical reference for effective management of refractory cases.


Diagnostic Methods: Physical examination revealed tenderness in the neck, chest, and lumbosacral region, along with pustular rashes and pruritus on both hands and feet. Laboratory testing showed negative Human Leukocyte Antigen B27 (HLA-B27). Computed tomography (CT) of the sacroiliac joints suggested sacroiliitis, and bone scan indicated costosternal joint involvement. SAPHO syndrome was confirmed based on these typical clinical manifestations.

Treatment Protocol: The patient underwent four sequential treatment stages. First, he received Etanercept combined with Sulfasalazine for 8 weeks. Due to suboptimal skin response, the regimen was changed to Secukinumab for another 8 weeks. When musculoskeletal symptoms recurred, treatment was adjusted to the JAK inhibitor Tofacitinib at 5 mg twice daily for 6 weeks. Finally, the regimen was further modified to incorporate Iguratimod at 25 mg twice daily combined with ongoing Tofacitinib for 6 weeks.


Following 8 weeks of Etanercept plus Sulfasalazine therapy, the patient's neck, chest, and back pain significantly improved, but the pustular rashes and itching on both hands and feet showed only slight improvement. After switching to Secukinumab for 8 weeks, the rashes on the hands and feet improved somewhat, but the neck, chest, and back pain recurred. With Tofacitinib monotherapy for 6 weeks, both the rashes and chest and back symptoms improved, though they did not fully resolve. After initiating the combination of Tofacitinib and Iguratimod for 6 weeks, the patient's chest, back, and rashes significantly improved. During the one-year follow-up period, the patient's condition remained stable with no signs of recurrence or disease progression.

This case represents the first reported successful treatment of SAPHO syndrome using the combination of the JAK inhibitor Tofacitinib and Iguratimod, offering a novel therapeutic option for this rare condition. 

关键词: SAPHO syndrome; treatment; JAK inhibitor; Iguratimod
来源:中华医学会第二十八次风湿病学学术会议