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Ruoyi Wang,1,2,* Yingzi Li,1,2,* Yuyue Liu,3 Xiujuan Hou,1,2,* Chen Li4,* 1Beijing University of Chinese Medicine, Beijing, People’s Republic of China; 2Department of Rheumatology, Dongfang Hospital Beijing University of Chinese Medicine, Beijing, People’s Republic of China; 3Department of Pathology, Dongfang Hospital Beijing University of Chinese Medicine, Beijing, People’s Republic of China; 4Department of Rheumatology, Fangshan Hospital Beijing University of Chinese Medicine, Beijing, People’s Republic of China*These authors contributed equally to this workCorrespondence: Xiujuan Hou; Chen Li, Email houxiujuan2008@163.com; casio1981@163.comIntroduction: SAPHO (synovitis, acne, pustulosis, hyperostosis, and osteitis) syndrome is a rare disease clinically characterized by a wide range of cutaneous and osteoarticular manifestations, involving multiple system impairments. Vasculitis is a rare comorbidity of SAPHO. Henoch–Schönlein purpura (HSP) is a vasculitis involving the capillaries and arterioles mediated by IgA immune complex. No case report of SAPHO syndrome with HSP was ever found.Case: Here we reported a case of SAPHO syndrome complicated with HSP and was successfully treated by methylprednisolone and tofacitinib.Discussion: Although the treat-to-target management of HSP and the first-line clinical medication have given some advices on the treatment. A precise treatment was still needed based on the pathogenesis of the comorbidity. The mechanism of the co-occurrence includes innate immunity and adapted immunity. Considering the active inflammatory reaction and the rapid disease progression, methylprednisolone and tofacitinib were prescribed.Conclusion: HSP is a new comorbidity of SAPHO. The spectrum of cutaneous small-vessel vasculitis in SAPHO syndrome was enriched. A new treatment approach for SAPHO with HSP was provided.Keywords: Henoch–Schönlein purpura, SAPHO syndrome, tofacitinib, vasculitis |