Long-term Evaluation of Mandibular Reconstruction with Fibular Flap in SAPHO Syndrome: 18 Years Post-onset.

Synovitis, acne, pustulosis, hyperostosis, and osteitis (SAPHO) syndrome is a systemic inflammatory disease that involves mandibular osteomyelitis with diffuse sclerosis in 10% of cases. Drug therapy is the primary treatment choice; however, severe deformity resulting in restricted mouth opening requires mandibular reconstruction. We report a rare case of mandible reconstruction in a patient with SAPHO syndrome, using a vascularized fibular flap with long-term follow-up. One year after surgery, a fistula developed in the chin region following a rash on both lower extremities as a skin manifestation of SAPHO syndrome. Bone scintigraphy showed uptake in the remaining right mandible, although there was no uptake in the transplanted fibula bone. Five years after the surgery, the mandibular fistula was closed by removing the titanium plate. The transplanted vascularized fibula achieved osteosynthesis; however, further follow-up is required to identify early symptoms of relapse and determine the need for additional medications.