Case reportLethal cervical abscess following bisphosphonate related osteonecrosis of the jaw
Introduction
Bisphosphonates have been used since the 1960s, mainly for the prevention and treatment of osteoporosis. Other common indications are Pagets' disease, multiple myeloma, bone metastases, and calcium disorders. Increased use of bisphosphonates has occurred during the past decade. These substances are currently considered to be the most frequently prescribed drugs for the treatment of osteoporosis (2006). An estimated 22 million prescriptions of alendronate were issued from March 2003 to April 2004 in the USA (2006).
Plotkin et al. discovered that the osteoclast-inhibiting effect of bisphosphonates is enhanced by additional reduction in apoptosis of osteoblasts and osteocytes (Bellido and Plotkin, 2011, Plotkin et al., 1999). Along with the increased clinical application of bisphosphonates, the first scientific reports about their complications were published. In 1996, de Groen described esophagitis as a common adverse effect of alendronate (de Groen et al., 1996). In a report of 36 bony lesions of the upper and lower jaws, bisphosphonate-related osteonecrosis of the jaw (BRONJ) was first described in 2003 by Marx et al. (Marx, 2003). Subsequent reports confirmed these findings to be an adverse effect of bisphosphonate therapy (Chiu et al., 2013, Melo and Obeid, 2005, Ruggiero et al., 2004).
In 2008, the Committee for Medicinal Products for Human Use (CHMP) defined the “osteonecrosis of the jaw related to bisphosphonates” as follows: (1) necrotic bone exposure in the maxillofacial region that has persisted for more than 8 weeks, (2) no history of irritation of the jaw, and (3) current or previous treatment with bisphosphonates. In addition, the CHMP proposed a staging system for BRONJ. While stages 1 and 2 are characterized only by the exposure or prevalence of necrotic bone with or without infection, stage 3 describes possible complications due to BRONJ (Use, 2009). Many of these complications are common, such as pathologic fractures, extra-oral fistula or osteolysis extending to the sinus floor, while others are rare. The present paper reports an unusual complication of BRONJ: bacterial embolism of the internal jugular vein with consecutive bacterial sepsis resulting in multi-organ failure and finally death after recurrent BRONJ-induced abscesses. To the best of the authors' knowledge, this complication of osteoporosis therapy with bisphosphonates has not been reported previously.
The aim of this paper is to report a rare case of bacterial embolism of the internal jugular vein after a BRONJ-induced submandibular abscess resulting in bacterial sepsis, multi-organ failure syndrome, and finally death.
Section snippets
Case report
A 59-year-old female patient presented herself to the Department for Cranio-Maxillofacial Surgery of the University Hospital Giessen, Germany with recent onset of a right submandibular swelling after tooth extraction at her family dentist. Suffering from compression fractures of the lumbar spine, the patient reported treatment with an oral bisphosphonate (alendronate 70 mg weekly) about 9 months previously for a six-month period for the treatment of osteoporosis, which had been stopped due to
Discussion
Management of BRONJ is a critical challenge in oral and maxillofacial surgery. Despite the reported cases of complications and therapeutic approaches, no definite cure has been identified (Favia et al., 2009, Wongchuensoontorn et al., 2009). Because of the successful outcomes of bisphosphonate treatment for the progression of metastatic cancer, prevention of fracture, and reduction of bone pain in osteoporosis therapy, interruption of the medication is only recommended when the risk of skeletal
Conclusion
Treatment options and diagnostic tests for risk stratification have been developed. However, careful instruction regarding oral hygiene practices, the importance of continuous follow-up, and the possible risks of BRONJ are recommended in the literature. To establish a definitive treatment for BRONJ, further epidemiological analysis of its characteristics, clinical trials and research on its pathogenesis are necessary to prevent cases similar to that described in this paper.
Disclosure statement
The authors have nothing to disclose.
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