Objective: To describe a unique case of unusually long treatment by oral Bisphosphonates associated with unresolving, treatment- resistant jaw and maxillary sinus complications.
Method: Retrospective analysis of disease course and microscopic evaluation.
Results: A 63 year old female first presented with swelling, pain, suppuration and bone exposure in the left maxilla. Extraction of both pre-molars had been performed 2 years previously, and the area failed to heal. Panoramic radiograph and CT scan showed opacification of the maxillary sinus, with increased density of the maxillary bone and adjacent sinus wall. The patient had continued to use oral Bisphosphonates, originally prescribed for osteoporosis, for an unusually long period of 17 years. A biopsy identified osteomyelitis associated with actinomycosis, compatible with BROJ. In spite of intensive treatment following the recommendations in the literature (drug-cessation for over 1 year, repeated and prolonged treatment by antibiotics, local rinses with chlorhexidine, removal of loose sequesters), pain and inflammation continued throughout this period and healing could not be achieved for more than 1 year of intensive treatment and 3 years since the extractions.
Conclusion: Although oral Bisphosphonates are only rarely associated with BROJ, lesions have been described to heal after discontinuation of the drug and conservative local control. The treatment-resistant osteomyelitis in the present case is most probably related to the extremely long oral Bisphosphonate treatment, and should serve as an indication of the potential danger for serious complications, with a devastating effect on patient's well being and quality of life.