Infliximab for Refractory Diffuse Sclerosing Osteomyelitis of the Mandible Associated With Ulcerative Colitis: A Case Report.
Yao Mayumi M, Obata Kyoichi K, Takei Kensuke K, Miyake Yoshitaka Y et al.
Diffuse sclerosing osteomyelitis (DSO) of the mandible is a chronic nonbacterial inflammatory bone disease. We report a 65-year-old woman with ulcerative colitis who presented with swelling and pain of the left mandible. The lesion was refractory to tooth extraction, prolonged antibiotics, and nonsteroidal anti-inflammatory drugs. CT showed widespread loss of trabecular structure and osteosclerosis, and bone scintigraphy demonstrated abnormal uptake confined to the mandible, without findings suggestive of synovitis, acne, pustulosis, hyperostosis, and osteitis syndrome. DSO was diagnosed based on the overall clinical course, progressive radiological findings, persistence despite treatment of the suspected odontogenic foci and repeated antibiotic therapy, and supportive histopathological findings. Anti-tumor necrosis factor-α (TNF-α) therapy was considered for the refractory DSO, and infliximab was selected because it is also effective for ulcerative colitis; her symptoms resolved promptly, and imaging showed marked improvement with restoration of trabecular bone. No relapse or treatment-related adverse events occurred during 3 years of follow-up. The clinical and radiographic improvement observed following infliximab treatment in this older adult raises the possibility that TNF-α-mediated inflammatory pathways may be involved in refractory DSO associated with ulcerative colitis and supports further investigation of anti-TNF-α therapy in this setting.