Abstract
Multiple myeloma is characterized by a neoplastic proliferation of plasma cells primarily in the bone marrow. Neoplastic plasma cells stimulated osteoclasts, and destroy bone tissue, causing bone pain, pathological fractures, paralysis due to spinal cord compression, and hypercalcemia. Bisphosphonates are used as supportive therapy in the management of multiple myeloma. Medication-related osteonecrosis of the jaw (MRONJ) is a well-known complication of treatment with bisphosphonates, denosumab, and other drugs, such as anti-angiogenic agents and novel anti-cancer drugs. We report MRONJ in a patient with multiple myeloma, especially an unusual case with tumor in the surgical specimen. A 73-year-old woman presented with pain on the left side of the mandible within 3 months. On clinical examination, an exposed bone without purulent drainage presented on the left side of the mandible. Before 2 years, she received chemotherapy of zoledronate for multiple myeloma at another hospital. Panoramic imaging showed radiopacities of bone in the left side of the mandibular molar area. Multidetector computed tomography (MDCT) with axial, multiplanar reformation (MPR) and three-dimensional (3D) images showed the sequestrum without periosteal reaction. She was diagnosed as MRONJ, and underwent surgery. Finally, the surgical specimen was diagnosed as multiple myeloma in the sequestrum. This case suggests that the evaluation of the surgical specimen of MRONJ could be essential for detection of primary tumor.
Similar content being viewed by others
References
Choi WS, Lee JI, Yoon HJ, Min CK, Lee SH. Medication-related osteonecrosis of the jaw: a preliminary retrospective study of 130 patients with multiple myeloma. Maxillofac Plast Reconstr Surg. 2017;39:1.
Auzina D, Slaidina A, Sevastjanova M, Erts R, Lejnieks A, Lejniece S. A population based study of multiple myeloma patients with medication-related osteonecrosis of the jaw. Stomatologija. 2019;21:13–7.
Ruggiero SL, Dodson TB, Fantasia J, Goodday R, Aghaloo T, Mehrotra B, 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;72:1938–56.
Fusco V, Cabras M, Erovigni F, Dell’Acqua A, Arduino PG, Pentenero M, et al. A multicenter observational study on medication-related osteonecrosis of the jaw (MRONJ) in advanced cancer and myeloma patients of a cancer network in North-Western Italy. Med Oral Patol Oral Cir Bucal. 2021;26:e466–73.
Bacci C, Cerrato A, Dotto V, Zambello R, Barilà G, Liço A, et al. The importance of alliance between hematologists and dentists: a retrospective study on the development of bisphosphonates osteonecrosis of the jaws (Bronj) in multiple myeloma patients. Dent J (Basel). 2021;9:11.
Ogura I, Kobayashi E, Nakahara K, Haga-Tsujimura M, Igarashi K, Katsumata A. Computer programme to assess mandibular cortex morphology in cases of medication-related osteonecrosis of the jaw with osteoporosis or bone metastases. Imaging Sci Dent. 2019;49:281–6.
Ogura I, Minami Y, Ono J, Kanri Y, Okada Y, Igarashi K, et al. CBCT imaging and histopathological characteristics of osteoradionecrosis and medication-related osteonecrosis of the jaw. Imaging Sci Dent. 2021;51:73–80.
Ogura I, Sasaki Y, Kameta A, Sue M, Oda T. Characteristic multimodal imaging of medication-related osteonecrosis of the jaw: comparison between oral and parenteral routes of medication administration. Pol J Radiol. 2017;82:551–60.
Ogura I, Sasaki Y, Sue M, Oda T, Kameta A, Hayama K. Tc-99m hydroxymethylene diphosphonate scintigraphy, computed tomography, and magnetic resonance imaging of osteonecrosis in the mandible: osteoradionecrosis versus medication-related osteonecrosis of the jaw. Imaging Sci Dent. 2019;49:53–8.
Ogura I, Kobayashi E, Nakahara K, Igarashi K, Haga-Tsujimura M, Toshima H. Quantitative SPECT/CT imaging for medication-related osteonecrosis of the jaw: a preliminary study using volume-based parameters, comparison with chronic osteomyelitis. Ann Nucl Med. 2019;33:776–82.
Ogura I, Sasaki Y, Sue M, Oda T, Kameta A, Hayama K. Tc-99m hydroxymethylene diphosphonate SPECT/CT for the evaluation of osteonecrosis of the jaw: preliminary study on diagnostic ability of maximum standardized uptake value. Clin Radiol. 2020;75:46–50.
Toshima H, Ogura I. Assessment of inflammatory jaw pathologies using bone SPECT/CT maximum standardized uptake value. Dentomaxillofac Radiol. 2020;49:20200043.
Minami Y, Ogura I. Bone single-photon emission computed tomography-CT peak standardized uptake value for chronic osteomyelitis, osteoradionecrosis and medication-related osteonecrosis of the jaw. J Med Imaging Radiat Oncol. 2021;65:160–5.
Ogawa R, Ogura I. Analysis of medication-related osteonecrosis of the jaw with bone SPECT/CT: relationship between patient characteristics and maximum standardized uptake value. Dentomaxillofac Radiol. 2021;50:20200516.
Vanpoecke J, Verstraete L, Smeets M, Ferri J, Nicot R, Politis C. Medication-related osteonecrosis of the jaw (MRONJ) stage III: conservative and conservative surgical approaches versus an aggressive surgical intervention: a systematic review. J Craniomaxillofac Surg. 2020;48:435–43.
Baba A, Goto TK, Ojiri H, Takagiwa M, Hiraga C, Okamura M, et al. CT imaging features of antiresorptive agent-related osteonecrosis of the jaw/medication-related osteonecrosis of the jaw. Dentomaxillofac Radiol. 2018;47:20170323.
Marx RE, Tursun R. Suppurative osteomyelitis, bisphosphonate induced osteonecrosis, osteoradionecrosis: a blinded histopathologic comparison and its implications for the mechanism of each disease. Int J Oral Maxillofac Surg. 2012;41:283–9.
Shuster A, Reiser V, Trejo L, Ianculovici C, Kleinman S, Kaplan I. Comparison of the histopathological characteristics of osteomyelitis, medication-related osteonecrosis of the jaw, and osteoradionecrosis. Int J Oral Maxillofac Surg. 2019;48:17–22.
Junquera S, Alvarez-Yague E, Junquera L, Ugalde R, Rua L. Multiple myeloma and chemical maxillary osteonecrosis. Can both occur simultaneously? J Stomatol Oral Maxillofac Surg. 2020;121:523–6.
Lu SY, Ma MC, Wang MC, Hsue SS. The status of jaw lesions and medication-related osteonecrosis of jaw in patients with multiple myeloma. J Formos Med Assoc. 2021;S0929–6646(21):00037–41.
Author information
Authors and Affiliations
Corresponding author
Ethics declarations
Conflict of interest
Ruri Ogawa, Yoshiyuki Minami, Junya Ono, Yoriaki Kanri, Eizaburo Kobayashi, Akira Tanaka, Yasuo Okada, and Ichiro Ogura declare that they have no conflict of interest.
Human rights statement
All procedures followed were in accordance with the ethical standards of the responsible committee on human experimentation (institutional and national) and with the Helsinki Declaration of 1975, as revised in 2008 (5).
Informed consent
Informed consent was obtained from all patients for being included in the study.
Additional information
Publisher's Note
Springer Nature remains neutral with regard to jurisdictional claims in published maps and institutional affiliations.
Rights and permissions
About this article
Cite this article
Ogawa, R., Minami, Y., Ono, J. et al. Medication-related osteonecrosis of the jaw in a patient with multiple myeloma: an unusual case with tumor in the surgical specimen. Oral Radiol 38, 288–291 (2022). https://doi.org/10.1007/s11282-021-00560-4
Received:
Accepted:
Published:
Issue Date:
DOI: https://doi.org/10.1007/s11282-021-00560-4