Osteonecrosis of the Jaw and Bilateral Atypical Femoral Fracture Both Occurring During Treatment for Osteoporosis: A Case Report

Authors

Keywords:

osteonecrosis of the jaw, subtrochanteric fracture, osteoporosis

Abstract

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.

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Author Biographies

Edelissa Payumo, Makati Medical Center, Makati City

Fellow-in-Training, Department of Medicine, Section of Endocrinology, Diabetes and Metabolism

Beinjerinck Ivan Cudal, Makati Medical Center, Makati City

Fellow-in-Training, Department of Medicine, Section of Endocrinology, Diabetes and Metabolism

Thelma Crisostomo, Makati Medical Center, Makati City

Consultant, Department of Medicine, Section of Endocrinology, Diabetes and Metabolism

References

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.

Published

2018-09-11

How to Cite

Payumo, E., Cudal, B. I., & Crisostomo, T. (2018). Osteonecrosis of the Jaw and Bilateral Atypical Femoral Fracture Both Occurring During Treatment for Osteoporosis: A Case Report. Journal of the ASEAN Federation of Endocrine Societies, 33(2), 194. Retrieved from https://www.asean-endocrinejournal.org/index.php/JAFES/article/view/469

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Section

Case Reports