Background/Purpose: Diffuse sclerosing osteomyelitis of the jaw is a rare and under-recognized disease. Many authors include this diagnosis in the spectrum of aseptic osteitis sometimes associated with the SAPHO syndrome. Differential diagnosis with infectious osteomyelitis is difficult, therefore the final diagnosis might be delayed by several years. Although considered as an auto-inflammatory disorder, patients can primarily be addressed to maxillofacial surgeons and not to the rheumatologist.

In this work, we describe a cohort of patients addressed to the maxillofacial surgery department for a spontaneous osteomyelitis of the jaw in order to identify characteristics that can help the surgeon to suspect this disease, make an earlier diagnosis and appropriate management.

Methods: We extracted patient records coded either “Osteomyelitis” or “Inflammatory conditions of jaws”(International Classification of Diseases) then selected the cases of spontaneous osteomyelitis of the mandible. Patients included had clinical and radiographic osteomyelitis for at least 3 months. Exclusion criteria were history of local radiotherapy, osteonecrosis and post-surgical infections. The final diagnosis was based on clinical and radiological outcomes at least one year after the first symptoms.

Results: Of the 960 records extracted, 24 patients met the inclusions criteria: 16 were classified as aseptic, 5 as infectious osteomyelitis and no definite diagnosis could finally be made for 3 of them. Aseptic osteomyelitis mainly affected women (10 out of 16) in their forties (median of 40.5 years) presenting with painful swelling of the mandible since several months (median of 8 months). Two patients presented an associated palmoplantar pustulosis leading to the diagnosis of SAPHO syndrome. Initial radiological examinations showed early osteolytic lesions (50%) progressing to medullary sclerosis, solid periosteal appositions and bone hypertrophy (90%).

Eleven out of 36 biopsies remained sterile but the others were positive for commensal germs of the oral flora (69%).

Interestingly, out of 8 patients who had a bone scintigraphy, 4 had other osteitis localizations allowing the diagnosis to be integrated into a SAPHO syndrome or chronic multifocal recurrent osteitis (CMRO).

Over 80% of patients received iterative antibiotic cures with inefficiency or recurrence of the pathology in all of them. Mandibular decortication was followed by 100% of recurrence. Among the 16 patients, 12 were finally treated with NSAIDs effectively on relapses, and 2 were cured with anti-TNF alpha.

Conclusion: Aseptic osteomyelitis remains rare but patients might be addressed directly to maxillofacial surgeons for management. It must be differentiated as early as possible from infectious osteomyelitis by its clinical and radiological characteristics. Germs of the oral flora can be found in the biopsy but might be only contamination. Most of patients of our series underwent multiple antibiotics cures and even surgery. We recommend that rheumatologist should be included as soon as possible to perform a clinical examination for extra-mandibular involvement to integrate aseptic osteomyelitis of the jaw into a SAPHO syndrome or CMRO and discuss NSAIDs or biologics.

ACR Meeting Abstracts - https://acrabstracts.org/abstract/diffuse-sclerosing-osteomyelitis-of-the-jaw-an-underdiagnosed-disease-in-maxillofacial-surgery-department/