TNF-alpha blockade in primary chronic non-bacterial osteomyelitis of the mandible

Abstract

Abstract Objectives Primary chronic non-bacterial osteomyelitis of the mandible (CNOM) is a rare auto-inflammatory disease of unknown aetiology that bears pathophysiological resemblance to both SAPHO syndrome in adults and chronic recurrent multifocal osteomyelitis (CRMO) in children. Both SAPHO and CRMO respond to TNF-α blockade. Previously reported treatment regimens in CNOM including NSAIDs, corticosteroids, antibiotics, anti-resorptive therapy and surgery all bear disappointing results. TNF-α blockade is suggested as a treatment option by some experts but this is not backed by any clinical data. We sought to retrospectively and exhaustively report our experience of anti-TNF-α therapy in refractory CNOM. Methods Fifteen patients with refractory CNOM and high disease burden were referred to our centre. TNF-α blockade was attempted in 10 cases, given its efficacy in neighbouring diseases, its good tolerance profile and failure of previous treatment strategies. We herein retrospectively report detailed outcomes for all patients having received anti-TNF-α therapy for this indication in our centre. Results TNF-α-targeting therapy resulted in a rapid and sustained remission in a majority of patients with CNOM, without serious adverse events. Treatment was tapered and stopped without relapse in some patients despite a refractory course of several years. Male sex seems to be associated with a poorer outcome. Conclusion Our results suggest that blocking TNF-α is efficient and safe in CNOM.