SAPHO syndrome complicated with relapsing polychondritis: A case report

Abstract

Synovitis, acne, pustulosis, hyperostosis, and osteitis (SAPHO) syndrome is a rare chronic inflammatory disease. The main clinical manifestation of SAPHO syndrome is an osteoarthropathy with cutaneous involvement. Relapsing polychondritis (RP) characterized by chronic inflammation and cartilage degeneration is a rare systematic autoimmune disease. Here we report a RP case in a SAPHO syndrome patient, in which auricularitis happened 10 years after the diagnosed as SAPHO syndrome. Tofacitinib treatment can alleviate the symptoms.

Tofacitinib: a successful treatment option for SAPHO syndrome

“…Only two reports evaluated the treatment response objectively by using scoring systems. 12,17 Third, the follow-up duration was relatively short for the majority of the cases (between 1.9 and 6 months) except for the case reported by Cao et al 15 and the present case (19 and 26 months, respectively). The considerably long follow-up periods of the latter reports are significant.…”

“…We reviewed 13 reports describing the treatment of SAPHO syndrome with tofacitinib in 36 patients (Table 1). [3][4][5][6][10][11][12][13][14][15][16] All patients had osteoarticular symptoms, and The literature review indicates that skin and joint manifestations of SAPHO can concurrently be treated with tofacitinib. However, there are some points to be noted.…”

Unusual cause of inflammatory backache: SAPHO syndrome

“…Furthermore, alendronate, a bisphosphonate prescribed at a weekly dosage of 70 mg, can provide significant and rapid relief of symptoms in patients with SAPHO syndrome. Bisphosphonates have a long‐term effect on inflammation and spinal bone marrow edema 18,19 . Initially, the patient exhibited a positive response to the treatment regimen.…”

JAKi: Can it be used to treat SAPHO syndrome?

“…Recently, a case of a patient who developed recurrent‐relapsing polychondritis (RP) 10 years after being diagnosed with SAPHO syndrome has been reported. The patient experienced relief of auricular chondritis with successful treatment with tofacitinib 19 . We are surprised to find that tofacitinib successfully treated SAPHO with Henoch–Schönlein purpura (HSP).…”