Exceptional response of skin symptoms to secukinumab treatment in a patient with SAPHO syndrome: Case report and literature review

Abstract

Rationale: SAPHO syndrome is a rare clinical entity characterized by a wide range of dermatological and musculoskeletal manifestations. Treatment strategies are not standardized. Palmoplantar pustulosis (PPP) is the most common rash in patients with SAPHO syndrome.Patient concerns: A 24-year-old Chinese woman with no relevant medical or familial history had a 1-year history of cutaneous lesions with PPP and pain in the sternoclavicular joint.Diagnosis: Based on the diagnostic criteria for SAPHO syndrome proposed by Nguyen et al in 2012, we diagnosed SAPHO syndrome with severe PPP as the predominant manifestation.Interventions: Due to the limited therapeutic efficacy of methotrexate and cyclosporin, we started therapy with subcutaneous secukinumab 150 mg weekly for the first month, then 150 mg monthly thereafter.Outcomes: After 4 weeks of secukinumab administration, the patient showed significant remission of pustular skin lesions, with almost no joint pain and no adverse reaction. Complete remission of skin symptoms was achieved after 3 months. Joint pain and adverse events have not reoccurred in follow-up thus far.Conclusions: In patients with SAPHO syndrome, we recommend personalized treatment, which may have excellent therapeutic efficacy in those with PPP or severe skin symptoms. Although data related to the use of IL-17 blockers for SAPHO syndrome are very limited, secukinumab provides a novel therapeutic option, especially for patients with PPP and severe skin lesions. Further prospective studies are needed to support our findings.

Successful Treatment of Refractory Synovitis, Acne, Pustulosis, Hyperostosis, and Osteitis (SAPHO) Syndrome and Paradoxical Psoriasis with Secukinumab: A Case Report

“…During the 7-month follow-up, her symptoms had resolved completely without complications or adverse reactions. 16 Sun et al described a male patient with SAPHO syndrome who presented with significant left jaw pain and mouth opening limitation. Despite undergoing successive treatments with pamidronate, tofacitinib, and adalimumab without achieving long-term remission, significant improvement in symptoms and remarkable remission on MRI were observed after treatment with secukinumab.…”

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

“…However, a variety of regimens, including glucocorticoids, bisphosphonates, some traditional antirheumatic drugs (such as methotrexate and leflunomide), and traditional Chinese medicines, have been reported to be effective in curbing disease deterioration and improving symptoms in SAPHO patients [ 21 ]. Both biological agents and JAK inhibitors have also achieved satisfactory remission, especially in patients with significantly elevated inflammatory levels after ineffective conventional drug treatment [ 6 , 7 , 11 ]. Since Propionibacterium acnes is a potential trigger for SAPHO syndrome, antibiotics may be an efficacious option for SAPHO syndrome treatment [ 22 ].…”

“…Secukinumab is a fully human monoclonal antibody targeting IL-17A which has been approved for the treatment of autoimmune psoriasis, psoriatic arthritis (PsA), and AS. Recently, secukinomab has been emerging as a promising treatment strategy for refractory SAPHO syndrome [ 6 , 7 ]. In the present case, we reported an extremely rare case of SAPHO syndrome complicated with AS with successful and complete resolution after treatment with secukinumab.…”

Percutaneous vertebroplasty for SAPHO syndrome with vertebral destruction: a case report and literature review

“…Palmoplantar pustules also resolved after 4 weeks. Ji et al ( 21 ) reported a 24-year-old woman who had a 1-year history of cutaneous manifestations with palmoplantar pustules and pain in the sternoclavicular joint. Methotrexate and cyclosporin had poor therapeutic effects.…”