Initial results of the treatment of diffuse sclerosing osteomyelitis of the mandible with bisphosphonates

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Abstract

Objective

Diffuse sclerosing osteomyelitis (DSO) of the mandible is characterised by recurrent pain, swelling of the cheek and trismus. Treatment is difficult and symptoms often recur. Recently case reports and small series have reported good results after treatment with bisphosphonates.

Study design

Seven patients suffering from treatment resistant DSO were treated with intravenous bisphosphonates. Diagnosis was based on clinical, radiological and histopathological examination. Symptoms had been present for between 20 and 167 months (mean 78). Patients were admitted, laboratory tests and Technetium-scans were performed. Pamidronate was administered intravenously up to four times a year. Follow-up varied from 18 to 46 months (mean 30).

Results

In all patients, symptoms and the need for analgesic drugs diminished considerably. One patient remained free of symptoms after one treatment. In two patients a switch in bisphosphonate was made based on a decreased response. All patients showed a decrease in uptake in the DSO-area on the Tc-scans when comparing pre- and post-treatment. One patient was lost to follow-up.

Conclusion

In therapy-resistant DSO bisphosphonate treatment may be a good option.

Introduction

Chronic diffuse sclerosing osteomyelitis (DSO) is characterised by recurrent pain, swelling of the cheek and trismus, without suppuration or formation of sequestra (Merkesteyn et al., 1988, Merkesteyn et al., 1990). Panoramic radiographs (Fig. 1) and CT-scans show areas of sclerosis and occasionally osteolysis, and Technetium-scans (Tc-scans) show increased uptake in the area of the lesion due to increased bone turnover.

The aetiology of DSO remains controversial. Some authors believe it is a response to a microbial stimulus (Jacobsson, 1984). Others have introduced chronic periostitis as a new concept of its aetiology (Merkesteyn et al., 1988, Merkesteyn et al., 1990, Groot et al., 1992a, Groot et al., 1992b).

The management of DSO can be quite challenging. Several strategies are often used, including long-term analgesic medication, antibiotics, and surgical interventions, for example decortication. In most cases these treatments do not succeed in achieving a long-lasting reduction of the recurrent pain, trismus and swelling. Some patients benefit from corticosteroids, but often this is only a temporary improvement.

Since the beginning of the current century several articles have been published concerning the treatment of DSO with bisphosphonates (Montonen et al., 2001, Soubrier et al., 2001, Sugata et al., 2003, Kopterides et al., 2004, Hino et al., 2005, Compeyrot-Lacassagne et al., 2007, Yamazaki et al., 2007). In most cases the bisphosphonate (pamidronate, clodronate or zoledronate) was given once. Follow-up extended to a maximum of 2 years. A remarkable decrease in complaints was noted in most patients. However, most articles are case reports. In this article a series of seven patients are reported.

Section snippets

Methods

Between March 2005 and December 2008 seven patients with a diagnosis DSO of the mandible, seen in the Departments of Oral and Maxillofacial Surgery and Endocrinology of the Leiden University Medical Centre, Leiden (LUMC), The Netherlands, were treated with the bisphosphonate pamidronate. Six patients were females, one was male. Ages varied from 12 to 78 years (Table 1). The diagnosis was based on the history, clinical symptoms, radiological and histopathological examination. Panoramic

Results

All patients reported a decrease in pain 1 or 2 days after the first infusion of pamidronate. Three patients no longer required analgesia at discharge, in the other four patients the need for analgesic drugs was considerably less (Table 2). Symptoms of trismus and swelling also diminished, although only briefly in some patients. During their hospital stay no patients developed fever or other flue-like symptoms.

Laboratory results showed no pre- or post-treatment abnormalities in their parameters

Discussion

Treatment of patients with DSO of the mandible is still challenging. Often symptoms have been present for a long time, and patients frequently undergo treatments that lead to discomfort without prolonged effects on their complaints. Surgical procedures such as decortications and resections with reconstructions have been performed in several patients, even repeatedly, with varying and often disappointing results (Montonen et al., 1993). Hyperbaric oxygen regimens have been tried, as well as

Conclusion

In DSO-patients that show recurrent disease after various treatments, treatment with bisphosphonates can lead to long-lasting relief of symptoms. Further research is needed for fine-tuning of the treatment.

Conflict of interest

There are no financial or personal relationships with other people or organisations that can inappropriately influence the work of the authors.

References (22)

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