Department of Rheumatology, CHU UCL Namur, Mont-Godinne, 1 Avenue Gaston Therasse, 5530, Yvoir, Belgium.
Department of Radiology, CHU UCL Namur, Mont-Godinne, 1 Avenue Gaston Therasse, 5530, Yvoir, Belgium.
J Med Case Rep. 2024 Feb 16;18(1):57. doi: 10.1186/s13256-024-04366-w.
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.
滑膜炎、痤疮、脓疱病、骨肥厚、骨炎（SAPHO）综合征是一种罕见病症。其治疗仍然是临床医生面临的挑战，并且往往效果不一。
我们报告了一例 51 岁白人女性 SAPHO 综合征病例，主要为轴性受累。她曾多年接受柳氮磺胺吡啶和抗炎药物治疗，但均未见效。开始使用托法替尼治疗数周后，临床和生物学参数均显著改善。影像学也显示脊柱和骨盆病变有明显消退。然而，由于发生肺栓塞，不得不停用托法替尼。因此，骨痛和生物炎症迅速复发。
抗 JAK 药物是治疗 SAPHO 综合征的一种有前途的治疗选择，需要进一步的临床试验和评估来验证疗效。
