Purpose: To report a case of bilateral retinal vasculitis successfully managed with adalimumab in a patient with synovitis, acne, pustulosis, hyperostosis, and osteitis (SAPHO) syndrome.
Case report: SAPHO syndrome was diagnosed in a 48-year-old female with bilateral blurred vision refractory to steroid eye drops. A preliminary ophthalmic examination revealed bilateral intermediate uveitis with vitreous opacity, and fluorescein angiography (FA) revealed dye leakage from peripheral retinal vessels. Because oral antirheumatic drugs were ineffective in treating her osteitis, her internist prescribed adalimumab, which resulted in rapid normalization of C-reactive protein and improvement of osteitis. Following treatment with adalimumab for 5 months, FA revealed significant improvement of retinal vasculitis. This is the first report on the use of adalimumab for retinal vasculitis associated with SAPHO syndrome.
Conclusion: We described a rare case of retinal vasculitis associated with SAPHO syndrome. Adalimumab treatment was effective in treating both osteitis and retinal vasculitis.
Keywords: Adalimumab; SAPHO syndrome; anti-tumor necrosis factor; osteitis; retinal vasculitis.