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Synovitis, Acne, Pustulosis, Hyperostosis and Osteitis (SAPHO) Syndrome with Henoch–Schönlein Purpura: A Case Report
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...
Autores principales: | , , , , |
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Formato: | Online Artículo Texto |
Lenguaje: | English |
Publicado: |
Dove
2023
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Materias: | |
Acceso en línea: | https://www.ncbi.nlm.nih.gov/pmc/articles/PMC10146069/ https://www.ncbi.nlm.nih.gov/pubmed/37123621 http://dx.doi.org/10.2147/CCID.S392909 |
Sumario: | 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. |
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