Bisphosphonate-associated osteonecrosis of the auditory canal An association between bisphosphonate exposure and man- dibular and maxillary osteonecrosis has recently been des- cribed (Marx, 2003; Ruggiero et al, 2004). We present the first reported case of bisphosphonate-associated osteonecrosis occurring outside the oral cavity. The patient was a 64-year-old man with a 9-year history of immunoglobulin G (IgG) kappa multiple myeloma, for which he had received an autologous stem cell transplant. His maintenance therapy initially comprised interferon alpha with pamidronate 90 mg monthly. With disease progression after 5 years, zoledronate 4 mg monthly was substituted and dexamethasone 40 mg/d 4 d each month with lenalidomide 15 mg/d were added. During this time, his clinical course was complicated by the development of bilateral idiopathic exostoses of the external auditory canals, with recurrent superinfection, and by loosen- ing of the upper right second molar and lower right second molar. The loose teeth were extracted and the left sided exostosis was removed without complication; no intervention was made on the right ear. He subsequently presented with painful, non-healing sockets at the site of the tooth extractions. Examination revealed ulcerated mucosa and exposed devitalised bone at these sites. Computed tomography showed an area of periodontal necro- sis, with formation of a sequestrum. There was no clinical or radiographic evidence of myeloma deposition or infection. No tissue biopsy was performed because of concerns regarding healing. Six months later, routine examination revealed a new area of painless ulceration of the left auditory canal at the site of the previous surgery. Necrotic bone was exposed, extending beyond the margins of ulceration. There had been no recent infection or trauma. Computed tomography and gallium- labelled white cell scanning confirmed an area of necrotic bone at this site. There was, again, no clinical or radiographic evidence of myeloma deposition or infection. In view of these findings, a diagnosis of bisphosphonate-associated osteonec- rosis of the jaw and auditory canal was made. The lesions improved with cessation of the bisphosphonate, local debri- dement and oral antibiotics. Further investigation of the pathogenesis and incidence of bisphosphonate-associated osteonecrosis is warranted. We postulate that the association between the development of this condition and trauma resulted from a reduced ability of bone to respond to physiological demands in the presence of bisphosphonate-induced reduced osseous remodelling and blood flow (Wood et al, 2002). While the oral cavity, with its high local bacterial load and probability of exposure of bone to the environment, is particularly susceptible, the present case demonstrates that this phenomenon may be generalised. We suggest that clinicians maintain a high index of suspicion for this condition in patients receiving long-term bisphosphonates and that trauma to bone at any site, including surgery, be avoided where possible. Mark N. Polizzotto, 1 Vincent Cousins 2 Anthony P. Schwarer 1 Departments of 1 Haematology and 2 Surgery, The Alfred Hospital, Melbourne, Australia. E-mail: a.schwarer@alfred.org.au References Marx R.E. (2003) Pamidronate (Aredia) and zoledronate (Zometa) induced avascular necrosis of the jaws: a growing epidemic. Journal of Oral and Maxillofacial Surgery, 61, 1115–1117. Ruggiero S.L., Mehrotra B., Rosenberg T.J. & Engroff S.L. (2004) Osteonecrosis of the jaws associated with the use of bispho- sphonates: a review of 63 cases. Journal of Oral and Maxillofacial Surgery, 62, 527–534. Wood J., Bonjean K., Ruetz S., Bellahcene A., Devy L., Foidart J.M., Castronovo V. & Green J.R. (2002) Novel antiangiogenic effects of the bisphosphonate compound zoledronic acid. Journal of Phar- macology and Experimental Therapy, 302, 1055–1061. Keywords: multiple myeloma, bisphosphonates. correspondence doi:10.1111/j.1365-2141.2005.05833.x ª 2005 Blackwell Publishing Ltd, British Journal of Haematology, 132, 114–117