Department of Rheumatism and Immunology, Ningbo No. 6 Hospital, Ningbo, China.
Department of Pharmacy, Ningbo No. 6 Hospital, Ningbo, China.
Front Immunol. 2022 May 25;13:911922. doi: 10.3389/fimmu.2022.911922. eCollection 2022.
Synovitis, acne, pustulosis, hyperostosis, and osteitis (SAPHO) syndrome, a type of chronic inflammatory disease, is rare and difficult to treat. Osteoarthropathy with skin involvement is the primary clinical manifestation of SAPHO syndrome. The unknown pathogenesis of SAPHO syndrome is speculated to be related to individual genetic differences, immune levels, microorganisms, and environmental factors. Tofacitinib, a novel small-molecule Janus kinase (JAK) inhibitor, has been used to treat rheumatoid arthritis. However, it also has great potential for the treatment of other immune diseases, including SAPHO syndrome. A 36-year-old man with chest and back pain for more than two months was admitted to our hospital. After admission, the patient developed a pustular rash and enteritis. SAPHO syndrome was diagnosed based on the above clinical manifestations, computed tomography (CT), and bone scintigraphy findings. Notably, the patient also had ankylosing spondylitis. Tofacitinib significantly improved the patient's skin symptoms while preventing worsening of chest and back pain when adalimumab was discontinued. We report the first case of ankylosing spondylitis with SAPHO syndrome. In addition, it is also the first successful treatment thereof with tofacitinib. We hope to provide valuable information regarding the pathogenesis and treatment of SAPHO syndrome in this case.
滑膜炎-痤疮-脓疱病-骨肥厚-骨炎(SAPHO)综合征是一种罕见且难以治疗的慢性炎症性疾病。伴有皮肤受累的骨关节炎是 SAPHO 综合征的主要临床特征。SAPHO 综合征的发病机制尚不清楚,据推测与个体遗传差异、免疫水平、微生物和环境因素有关。托法替布是一种新型小分子 Janus 激酶(JAK)抑制剂,已被用于治疗类风湿关节炎。然而,它在治疗其他免疫疾病方面也具有很大的潜力,包括 SAPHO 综合征。一名 36 岁男性,因胸痛和背痛超过两个月而入院。入院后,患者出现脓疱性皮炎和肠炎。根据上述临床表现、计算机断层扫描(CT)和骨闪烁显像结果,诊断为 SAPHO 综合征。值得注意的是,该患者还患有强直性脊柱炎。托法替布在停用阿达木单抗后,显著改善了患者的皮肤症状,同时预防了胸痛和背痛的恶化。我们报告了首例强直性脊柱炎伴 SAPHO 综合征的病例。此外,这也是首例使用托法替布成功治疗的病例。我们希望在该病例中为 SAPHO 综合征的发病机制和治疗提供有价值的信息。