Osteonecrosis of the Jaw and Bilateral Atypical Femoral Fracture Both Occurring During Treatment for Osteoporosis: A Case Report
Osteonecrosis of the jaw (ONJ) and atypical femoral fracture (AFF) are rare potential adverse effects of bisphosphonates and RANKL antibody therapy. The pathogenic mechanisms of both conditions are known to be independent of each other. Here, we report both conditions sequentially occurring in the same patient.
An 81-year-old, obese, diabetic, female was admitted due to hypertensive urgency and persistent jaw pain after tooth extraction. The patient has postmenopausal osteoporosis for fourteen years and was on intermittent, unsupervised treatment with alendronate, denosumab and ibandronate. Upon presentation, the patient was noted with tenderness intraorally of tooth number 35 periapical region. This was associated with elevated erythrocyte sedimentation rate and C-reactive protein. Imaging study showed presence of bony sclerosis which represent a sequestrum in the molar area of the left hemi-mandible. Antibiotic infusion and excision and debridement of left posterior mandible were done. Histopathologic finding was consistent with a diagnosis of osteonecrosis of the jaw. The same patient, upon review, had suffered sequential fracture of both femurs during the eighth and eleventh year of treatment with antiresorptive agents. The fractures were transverse, non-comminuted, at the proximal femoral shaft. Each occurred after a minor trauma and was managed with open reduction and internal fixation. Both fractures were consistent with atypical femoral fractures.
ONJ and AFF can occur both in the same patient during prolonged treatment with bisphosphonates and denosumab and may suggest a common pathogenic mechanism.
Cosman F, De Beur SJ, LeBoff MS, Lewiecki EM, Tanner B, Randall S. Clinician’s guide to prevention and treatment of osteoporosis. Osteoporosis Int. 2014;25(10):2359-81. PMID: 25182228. PMCID: PMC4176573. https://doi.org/10.1007/s00198-014-2794-2.
Black DM, Schwartz AV, Ensrud KE, et al. Effects of continuing or stopping alendronate after 5 years of treatment. The Fracture Intervention Trial Long-term Extension (FLEX): A randomized trial. JAMA. 2006;296(24):2927-38. PMID: 17190893. https://doi.org/10.1001/jama.296.24.2927.
Cummings SR, San Martin J, McClung MR, et al. Denosumab for prevention of fractures in postmenopausal women with osteoporosis. N Engl J Med. 2009;361(8):756-65. PMID: 19671655. https://doi.org/10.1056/NEJMoa0809493.
Ruggiero SL, Dodson TB, Fantasia J, et al. American Association of Oral and Maxillofacial Surgeons position paper on medication-related osteonecrosis of the jaw – 2014 update. J Oral Maxillofac Surg. 2014;72(10):1938-56. PMID: 25234529. https://doi.org/10.1016/j.joms.2014.04.031.
Shane E, Burr D, Abrahamsen B, Adler R, et al. Atypical subtrochanteric and diaphyseal femoral fractures: Second report of a Task Force of the American Society for Bone and Mineral Research. J Bone Miner Res. 2014; 29(1):1–23. PMID: 23712442. https://doi.org/10.1002/jbmr.1998.
Hu CJ, Renn JH, Yang SW, Lin KC. Atypical femoral fractures in a patient with continuous decreasing BMD after only 1.5 Years of bisphosphonate treatment. Open Journal of Rheumatology and Autoimmune Diseases. 2014;4(1), Article ID: 42344. https://doi.org/10.4236/ojra.2014.41004.
Wąsowski M and Tałałaj M. Osteonecrosis of the jaw and atypical femoral fractures as complications of antiresorptive therapy. Post N Med 2017;XXX(1):43-8. http://www.pnmedycznych.pl/wp-content/uploads/2017/02/pnm_2017_043_048.pdf.
Peer A, Khamaisi M. Diabetes as a risk factor for medication-related osteonecrosis of the jaw. J Dent Res. 2015;94(2): 252-60. PMID: 25477311. PMCID: PMC4438733. https://doi.org/10.1177/0022034514560768.
Chiu WY, Lee JJ, Tsai KS. Atypical femoral fractures shortly after osteonecrosis of the jaw in a postmenopausal woman taking alendronate for osteoporosis. J Clin Endocrinol Metab 2013;98(4):E723-6. PMID: 23471975. https://doi.org/10.1210/jc.2012-4144.
Won Y, Lim JR, Kim YH, Song HK, Yang KH. Atypical femoral fracture combined with osteonecrosis of jaw during osteoporosis treatment with bisphosphonate. J Bone Metab. 2014;21(2):155-9. PMID: 25025002. PMCID: PMC4075270. https://doi.org/10.11005/jbm.2014.21.2.155.
Kim JE, Yun M, Lim SK, Rhee Y. Concurrent bisphosphonate-related bilateral atypical subtrochanteric fractures and osteonecrosis of the jaw on bone scintigraphy. Clin Nucl Med. 2015;40:450-2. PMID: 25706787. https://doi.org/10.1097/RLU.0000000000000746.
Sánchez A, Blanco R. Osteonecrosis of the jaw (ONJ) and atypical femoral fracture (AFF) in an osteoporotic patient chronically treated with bisphosphonates. Osteoporos Int. 2017;28(3):1145-7. PMID: 27866217. https://doi.org/10.1007/s00198-016-3840-z.
Copyright (c) 2018 Journal of the ASEAN Federation of Endocrine Societies
This work is licensed under a Creative Commons Attribution-NonCommercial 4.0 International License.
Journal of the ASEAN Federation of Endocrine Societies is licensed under a Creative Commons Attribution-NonCommercial 4.0 International. (full license at this link: http://creativecommons.org/licenses/by-nc/3.0/legalcode).
To obtain permission to translate/reproduce or download articles or use images FOR COMMERCIAL REUSE/BUSINESS PURPOSES from the Journal of the ASEAN Federation of Endocrine Societies, kindly fill in the Permission Request for Use of Copyrighted Material and return as PDF file to email@example.com or firstname.lastname@example.org.
A written agreement shall be emailed to the requester should permission be granted.