Yang Qiao, Zhao Yumo, Li Chen, Luo Yaping, Hao Weixin, Zhang Wen
Department of Traditional Chinese Medicine, Peking Union Medical College Hospital Peking Union Medical College Department of Nuclear Medicine, Peking Union Medical College Hospital Department of Rheumatology and Clinical Immunology, Peking Union Medical College Hospital, Chinese Academy of Medical Sciences and Peking Union Medical College, Beijing, PR China.
Medicine (Baltimore). 2018 Jun;97(25):e11149. doi: 10.1097/MD.0000000000011149.
Synovitis, acne, pustulosis, hyperostosis, and osteitis (SAPHO) syndrome is an autoinflammatory disorder without standardized treatment. Janus kinase (JAK) inhibitors can block a range of cytokines and might possess significant anti-inflammatory activity. Here, we report the first case of efficacious treatment of refractory SAPHO syndrome with the JAK inhibitor tofacitinib.
A 44-year-old woman presented with arthralgia in the right wrist and complained of having difficulty in doing housework. Symptoms were unresponsiveness to nonsteroidal anti-inflammatory drugs, disease-modifying antirheumatic drugs, and tumor necrosis factor inhibitors. A diagnosis of SAPHO syndrome was made based on previous dermatological and osteoarticular manifestations and bone scintigraphy findings. Oral treatment with tofacitinib at 5 mg twice daily in combination with the basic methotrexate treatment was initiated. After 4 weeks of using tofacitinib, the patient reported marked improvement of symptoms and also reported being competent in completing housework.
The efficacy of JAK inhibitors in treating refractory SAPHO syndrome should be noted.
滑膜炎、痤疮、脓疱病、骨肥厚和骨炎（SAPHO）综合征是一种缺乏标准化治疗的自身炎症性疾病。Janus激酶（JAK）抑制剂可阻断一系列细胞因子，可能具有显著的抗炎活性。在此，我们报告首例使用JAK抑制剂托法替布有效治疗难治性SAPHO综合征的病例。
一名44岁女性因右腕关节疼痛就诊，主诉做家务困难。症状对非甾体抗炎药、改善病情抗风湿药和肿瘤坏死因子抑制剂均无反应。根据既往皮肤和骨关节表现以及骨闪烁显像结果，诊断为SAPHO综合征。开始口服托法替布，每日两次，每次5毫克，并联合基本的甲氨蝶呤治疗。使用托法替布4周后，患者报告症状明显改善，还表示能够胜任家务。
应注意JAK抑制剂治疗难治性SAPHO综合征的疗效。
