Is primary chronic osteomyelitis a uniform disease? Proposal of a classification based on a retrospective analysis of patients treated in the past 30 years

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Abstract

Introduction: Primary chronic osteomyelitis of the jaw is a rare, non-suppurative, chronic inflammatory disease of unknown aetiology. To date, classification is confusing due to a non-uniform terminology. The aim of this study was to establish a simple (clinical) classification based on patient data from our clinic.

Methods: Retrospective analysis revealed 30 cases of which clinical course, radiology, pathology, therapy and outcome were analysed.

Results: Both sexes were equally represented. The mean age at onset of disease was 35 years (range 5–76 years). Onset of disease revealed two peaks of incidence, one in adolescence and one after age 50 years. While clinical symptoms were similar in all cases, an increased intensity of these symptoms was noted in younger individuals as well as in the early stages of the disease. Five adults and one adolescent presented with additional non facial bone, joint and skin manifestations consistent with the diagnosis of SAPHO (synovitis, acne, pustulosis, hyperostosis, osteitis) syndrome, or chronic recurrent multifocal osteomyelitis. Radiology demonstrated sclerosis, osteolysis and periosteal reaction in variable stages in all cases. However, findings were more extensive in younger patients. Histology revealed different stages of chronic inflammation in all cases. Microabscess formation was noted in 11 cases, six of which were children/adolescents.

Therapy consisted mainly of surgery, antibiotics and hyperbaric oxygen therapy.

At the end of the follow up period, 11 patients demonstrated complete remission, while in 14 cases amelioration and in 5 no significant improvement was noted.

Conclusion: Based on differences in age at presentation, clinical appearance and course, radiology and histology, a subclassification into early and adult onset primary chronic osteomyelitis has been established. Cases with purely mandibular involvement should further be distinguished from cases associated with other syndromes.

Section snippets

INTRODUCTION

Osteomyelitis is defined as an inflammatory process involving cortical and cancellous bone. In the maxillofacial region the mandible is by far the most frequently affected bone. In the vast majority of cases an odontogenic infectious aetiology is apparent. However, in some cases, no clear causal factor can be found.

An overview of the literature on osteomyelitis reveals a wide variety of proposed classifications based on different aspects such as clinical course, pathological-anatomical and/or

PATIENTS AND METHODS

Records from patients with primary chronic osteomyelitis from the last 30 years treated at the Department of Cranio-Maxillofacial Surgery, University Hospital Zürich were reviewed. Charts and radiographs were analysed retrospectively with special emphasis on demographic data, patient history, diagnostic parameters, treatment modalities, clinical course and outcome.

Thirty, well-documented cases were identified, with equal sex distribution. The mean age at onset was 35.0 years (range 5–75 years).

RESULTS

Age at onset of symptoms demonstrated two incidence peaks. In 13 cases, onset was noted before age 20, and in 9 patients after the age of 50 years (Table 1). Pain, local swelling and limited mouth opening were noticed as recurrent episodes and were the predominant clinical symptoms (Fig. 1). Regional lymphadenopathy and hyposensibility of the inferior alveolar nerve (Vincent's symptom) were noticed infrequently. Notably, 11 patients had symptoms consistent with a myofacial pain syndrome, such

DISCUSSION

Primary chronic osteomyelitis of the jaw is a poorly described disease entity. Whilst it has been reported in all age groups, most published data refers to adults (Jacobsson, 1984; Van Merkestyn et al., 1988; Montonen et al., 1993). Those scarce cases of primary chronic osteomyelitis with an onset in childhood and adolescence have mostly been presented as single case reports. (Panders and Hadders, 1970; Ellis et al., 1977; Eisenbud et al., 1981; Mattison et al., 1981; Nortje et al., 1988; Betts

CONCLUSION

Primary chronic osteomyelitis of the jaw is currently regarded a distinct disease entity, representing a chronic non-suppurative inflammation. However, the data presented in this study demonstrates that there were two main groups regarding the onset of symptoms: The first group with an early onset in adolescence, and a second group with a late onset peaking after the fifth decade. Considering clinical presentation and the course of the disease, primary chronic osteomyelitis seems to be more

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