Case report
Lethal cervical abscess following bisphosphonate related osteonecrosis of the jaw

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Abstract

Context

Bisphosphonates are common drugs used in the management of bone metabolic diseases. Because of their recently increased use, their adverse effects, especially bisphosphonate-related osteonecrosis of the jaw (BRONJ), are monitored more frequently. BRONJ is a critical challenge in craniofacial surgery and is difficult to treat. Its occurrence is either spontaneous or follows dentoalveolar surgery. Typical complications of BRONJ are painful exposed bone, pathological fractures, extra-oral fistula, and local infections.

Objective

The aim of this paper is to report a rare case of bacterial embolism in the internal jugular vein after a BRONJ-induced submandibular abscess resulting in bacterial sepsis, multi-organ failure syndrome, and death.

Case illustration

A 59-year-old female patient developed severe BRONJ (stage II) with recurrent abscesses after oral osteoporosis therapy with alendronic acid. A subsequent submandibular abscess led to bacterial embolism of the left internal jugular vein, causing sepsis and death.

Discussion

Prevention, early detection and management of BRONJ remain a crucial challenge in craniofacial clinical practice. Despite several therapeutic approaches described in the current literature, none have undergone bedside application.

Conclusion

Considering this report of death after recurrent abscesses following BRONJ, the use of bisphosphonates should be carefully monitored in order to prevent such severe complications.

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.

References (25)

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