Ferraioli Mario, Fiannacca Luigi, Greco Elisabetta, Cela Eneida, Fatica Mauro, Bergamini Alberto, Chimenti Maria Sole
Rheumatology, Allergology and Clinical Immunology, Department of Medicina dei Sistemi, University of Rome Tor Vergata, Rome, Italy.
Case Reports Immunol. 2024 Mar 19;2024:9076852. doi: 10.1155/2024/9076852. eCollection 2024.
SAPHO syndrome is a complex disease that encompasses both inflammatory arthritis and/or osteitis and dermatologic manifestations. It is considered a rare disease, in fact, no clinical trials have been conducted on its therapy and management. Therefore, therapeutic approach is based on small case studies. Here, we described the case of a 63-year-old woman affected by SAPHO syndrome, treated with the selective IL-23p19 antagonist, Risankizumab, after unsuccessful therapies with Methotrexate, Infliximab, Adalimumab, and an allergic reaction to Secukinumab. At the beginning of therapy, in November 2022, the patient presented with arthritis in both knees associated with palmar pustulosis and guttate psoriasis on the trunk. DAPSA score was 24, PtGA 80 mm, PASI score 11.1, and BSA 40%. Thereafter, Risankizumab was started at the standard dosage of 150 mg. At week 24 patient achieved clinical remission, DAPSA score was 8, PtGA was 30 mm, PASI was 1, and BSA 2.5. Patient maintained clinical remission state at the subsequent week 52 evaluation. At the same time, the patient did not report any adverse effects. Health-related quality of life was also assessed at the same time points aforementioned, showing significant improvement. In conclusion, this case report wants to point out the efficacy and safety of Risankizumab in SAPHO syndrome, reporting a sustained disease remission through a 12 months long follow-up period. We can consider IL-23p19 targeted therapy as a novel treatment option for SAPHO-with a high efficacy potential-especially on patients that have already been treated with other biologics.
滑膜炎、痤疮、脓疱病、骨肥厚和骨髓炎综合征（SAPHO综合征）是一种复杂疾病，包括炎性关节炎和/或骨炎以及皮肤表现。它被认为是一种罕见疾病，事实上，尚未针对其治疗和管理开展临床试验。因此，治疗方法基于小型病例研究。在此，我们描述了一名63岁患SAPHO综合征女性的病例，该患者在使用甲氨蝶呤、英夫利昔单抗、阿达木单抗治疗失败且对司库奇尤单抗出现过敏反应后，接受了选择性白细胞介素-23p19拮抗剂瑞莎珠单抗治疗。在2022年11月治疗开始时，患者双膝关节出现关节炎，伴有掌跖脓疱病和躯干点滴状银屑病。疾病活动度评分（DAPSA）为24分，患者总体评估（PtGA）为80毫米，银屑病面积和严重程度指数（PASI）评分为11.1，体表面积（BSA）为40%。此后，开始以150毫克的标准剂量使用瑞莎珠单抗。在第24周时患者实现临床缓解，DAPSA评分为8分，PtGA为30毫米，PASI为1分，BSA为2.5%。在随后第52周的评估中患者维持临床缓解状态。同时，患者未报告任何不良反应。在上述相同时间点还评估了健康相关生活质量，结果显示有显著改善。总之，本病例报告旨在指出瑞莎珠单抗治疗SAPHO综合征的有效性和安全性，报告了长达12个月随访期内疾病的持续缓解情况。我们可以将白细胞介素-23p19靶向治疗视为SAPHO综合征的一种新治疗选择——具有很高的疗效潜力——尤其是对于已经接受过其他生物制剂治疗的患者。
