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

Medicine (Baltimore). 2022 Aug 19;101(33):e30065. doi: 10.1097/MD.0000000000030065.

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.

Publication types

  • Case Reports
  • Review

MeSH terms

  • Acquired Hyperostosis Syndrome* / complications
  • Acquired Hyperostosis Syndrome* / diagnosis
  • Acquired Hyperostosis Syndrome* / drug therapy
  • Adult
  • Antibodies, Monoclonal, Humanized / therapeutic use
  • Female
  • Humans
  • Pain
  • Psoriasis*
  • Young Adult

Substances

  • Antibodies, Monoclonal, Humanized
  • secukinumab