Synovitis, Acne, Pustulosis, Hyperostosis and Osteitis (SAPHO) Syndrome with Henoch-Schönlein Purpura: A Case Report

Ruoyi Wang; Yingzi Li; Yuyue Liu; Xiujuan Hou; Chen Li

doi:10.2147/CCID.S392909

Synovitis, Acne, Pustulosis, Hyperostosis and Osteitis (SAPHO) Syndrome with Henoch-Schönlein Purpura: A Case Report

Clin Cosmet Investig Dermatol. 2023 Apr 24:16:1089-1094. doi: 10.2147/CCID.S392909. eCollection 2023.

Authors

Ruoyi Wang^#^{1

2}, Yingzi Li^#^{1

2}, Yuyue Liu³, Xiujuan Hou^#^{1

2}, Chen Li^#⁴

Affiliations

¹ Beijing University of Chinese Medicine, Beijing, People's Republic of China.
² Department of Rheumatology, Dongfang Hospital Beijing University of Chinese Medicine, Beijing, People's Republic of China.
³ Department of Pathology, Dongfang Hospital Beijing University of Chinese Medicine, Beijing, People's Republic of China.
⁴ Department of Rheumatology, Fangshan Hospital Beijing University of Chinese Medicine, Beijing, People's Republic of China.

^# Contributed equally.

Abstract

Introduction: 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.

Publication types

Case Reports

Grants and funding

This research was funded by the National Natural Science Foundation of China (No. 82074246) for CL and Construction Project of Clinical Key Specialty in Fengtai District of Beijing for XH.