Initial results of the treatment of diffuse sclerosing osteomyelitis of the mandible with bisphosphonates
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)
- et al.
“Bis-phossy jaws” – high and low risk factors for bisphosphonate-induced osteonecrosis of the jaw
J Craniomaxillofac Surg
(2008) - et al.
Treating low and median potent bisphosphonate-related osteonecrosis of the jaws with a protocol for the treatment of chronic suppurative osteomyelitis; report of 7 cases
Oral Surg Oral Med Oral Pathol Oral Radiol Endod
(2009) - et al.
Bisphosphonate-induced osteonecrosis of the jaws: prospective study of 80 patients with multiple myeloma and other malignancies
Oral Oncol
(2008) - et al.
Diffuse sclerosing osteomyelitis (chronic tendoperiostitis) of the mandible. An 11-year follow-up report
Oral Surg Oral Med Oral Pathol
(1992) - et al.
Changes in masseter inhibitory reflex responses in patients with diffuse sclerosing osteomyelitis of the mandible
Oral Surg Oral Med Oral Pathol
(1992) - et al.
The value of FDG–PET and bone scintigraphy with SPECT in the primary diagnosis and follow-up of patients with chronic osteomyelitis of the mandible
Int J Oral Maxillofac Surg
(2006) - et al.
Response of diffuse sclerosing osteomyelitis of the mandible to alendronate: follow-up study by 99mTc scintigraphy
Int J Oral Maxillofac Surg
(2005) - et al.
Treatment and prognosis of diffuse sclerosing osteomyelitis (DSO) of the mandible
Oral Surg Oral Med Oral Pathol
(1980) Diffuse sclerosing osteomyelitis of the mandible
Int J Oral Surg
(1984)- et al.
Treatment of chronic sclerosing osteomyelitis of the mandible with calcitonin: a report of two cases
Br J Oral Maxillofac Surg
(2005)
Decortication in the treatment of diffuse sclerosing osteomyelitis of the mandible. Retrospective analysis of 41 cases between 1969 and 1990
Oral Surg Oral Med Oral Pathol
Cited by (49)
Remarkable response of diffuse sclerosing osteomyelitis of the mandible to zoledronate by single infusion without prior treatment of other bisphosphonates
2022, Journal of Oral and Maxillofacial Surgery, Medicine, and PathologyBisphosphonate therapy in chronic diffuse sclerosing osteomyelitis/tendoperiostitis of the mandible: Retrospective case series
2022, Journal of Cranio-Maxillofacial SurgeryCitation Excerpt :Bisphosphonates can be administered orally or intravenously in different dosages. Two types of bisphosphonates are registered, nitrogen-containing (pamidronate, olpadronate, alendronate, ibandronate, zoledronic acid, risedronate) and non-nitrogen-containing (disodium clodronate) bisphosphonates (Montonen et al., 2001; Soubrier et al., 2001; Eyrich et al., 2003; Baltensperger et al., 2004; Hino et al., 2005; Compeyrot-Lacassagne et al., 2007; Kuijpers et al., 2011; Urade et al., 2012; Otto et al., 2015). Nitrogen-containing bisphosphonates are more common in the treatment for DSO/TP of the mandible, because they have a higher affinity for bone, a higher bioavailability and a possible longer duration of action than non-nitrogen-containing bisphosphonates (Otto et al., 2015; van de Meent et al., 2020).
Is Operative Management Effective for Non-Bacterial Diffuse Sclerosing Osteomyelitis of the Mandible?
2021, Journal of Oral and Maxillofacial SurgeryApplication of pamidronate disodium for the treatment of diffuse sclerosing osteomyelitis of the mandible: A clinical study
2020, Oral Surgery, Oral Medicine, Oral Pathology and Oral RadiologyDiffuse sclerosing osteomyelitis: a case series and literature review
2020, Oral Surgery, Oral Medicine, Oral Pathology and Oral Radiology