SAPHO Syndrome Complicated by Ankylosing Spondylitis Successfully Treated With Tofacitinib: A Case Report

Abstract

Synovitis, acne, pustulosis, hyperostosis, and osteitis (SAPHO) syndrome, a type of chronic inflammatory disease, is rare and difficult to treat. Osteoarthropathy with skin involvement is the primary clinical manifestation of SAPHO syndrome. The unknown pathogenesis of SAPHO syndrome is speculated to be related to individual genetic differences, immune levels, microorganisms, and environmental factors. Tofacitinib, a novel small-molecule Janus kinase (JAK) inhibitor, has been used to treat rheumatoid arthritis. However, it also has great potential for the treatment of other immune diseases, including SAPHO syndrome. A 36-year-old man with chest and back pain for more than two months was admitted to our hospital. After admission, the patient developed a pustular rash and enteritis. SAPHO syndrome was diagnosed based on the above clinical manifestations, computed tomography (CT), and bone scintigraphy findings. Notably, the patient also had ankylosing spondylitis. Tofacitinib significantly improved the patient’s skin symptoms while preventing worsening of chest and back pain when adalimumab was discontinued. We report the first case of ankylosing spondylitis with SAPHO syndrome. In addition, it is also the first successful treatment thereof with tofacitinib. We hope to provide valuable information regarding the pathogenesis and treatment of SAPHO syndrome in this case.

Successful Treatment of SAPHO Syndrome Complicated with Ankylosing Spondylitis by Secukinumab: A Case Report

“…Specifically, this case was complicated with AS. To date, only a few cases of SAPHO syndrome with AS have been reported, and the case’s skin lesions and osteoarticular pain were significantly improved by tofacitinib or secukinumab when adalimumab was discontinued [ 24 , 25 ]. Some scholars believe that SAPHO syndrome can be classified in the category of SpA, which is between AS and PsA [ 3 , 26 ].…”

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

“…It also has a convinced effect in the treatment of psoriatic arthritis, 23 ankylosing spondylitis, 24 and SAPHO syndrome. 9,25,26 Although tofacitinib was reported to treat refractory cutaneous leukocytoclastic vasculitis, it had not been reported in HSP treatment. Based on the inference of the co-occurrence mechanism of the two diseases and our previous experiences, tofacitinib was prescribed.…”

Novel JAK‐1 inhibitor upadacitinib as a possible treatment for refractory SAPHO syndrome: A case report

“…It has been reported that tofacitinib was effective in improving skin damage and reducing bone pain in a patient with ankylosing spondylitis combined with SAPHO syndrome. 5 In contrast, upadacitinib is a novel JAK1 inhibitor. Compared to the first-generation JAK inhibitor tofacitinib, upadacitinib can inhibit a single JAK protein with high selectivity without affecting other cytokines, thus potentially reducing adverse effects.…”