Maxillofacial Symptoms and Diagnosis Criteria Concerning Chronic Recurrent Multifocal Osteomyelitis: A Systematic Literature Review

Abstract

Introduction: Chronic recurrent multifocal osteomyelitis (CRMO) is a rare autoinflammatory disease characterized by sterile inflammation of bony tissue. Although it has distinct clinical and radiologic features, it is often misdiagnosed, leading to inappropriate and delayed treatment. Indeed, at the origin the disease can have an exclusive maxillofacial location and so be, at least clinically, difficult to prove. Objective: The aim of this study was to summarize through a systematic literature review the clinical, laboratory, and imaging data regarding maxillofacial manifestations of CRMO. Methods: The present systematic review of the literature was designed following the standard PROSPERO eligibility criteria. Three electronic databases were searched to find clinical studies describing cases of patients diagnosed with CRMO of the orofacial area. Relevant clinical, radiographic, and laboratory results were extracted. Results: The search strategy retrieved 95 articles. Assessment of the full text was done for 47 articles among them, 30 were included. The total patients included was 82, mean age was 8.2 years. Pain and swelling association was present in 94% of the cases, mandible was involved in 96.3% of the cases. Sclerosis, lytic lesion, and periostal reaction were the most frequent radiologic findings. Conclusion: Chronic recurrent multifocal osteomyelitis is often misdiagnosed as infectious osteomyelitis, practitioners should therefore be familiar of its distinct clinical course and paraclinical features. Further investigations would be beneficial to assess the extraosseous manifestations and the long-term outcome of CRMO.