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Published in: BMC Oral Health 1/2021

Open Access 01-12-2021 | Osteonecrosis | Case report

Mucormycosis originated total maxillary and cranial base osteonecrosis: a possible misdiagnosis to malignancy

Authors: Young Long Park, Sura Cho, Jin-Woo Kim

Published in: BMC Oral Health | Issue 1/2021

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Abstract

Background

This is a case of mucormycosis originated osteonecrosis of the maxilla extended to the cranial base, initially suspected of malignancy. The patient was first suspected with osteolytic sarcomatous lesion but was later diagnosed with total maxillary necrosis and cranial base through biopsy-proven invasive mucormycosis.

Case presentation

A 71-year-old male was presented with unknown total maxillary osteonecrosis. CT and MRI results showed extensive osteolytic change with bone destruction of the cranial base, and PET-CT showed irregular hypermetabolic lesion in the area suspected of malignancy. The first biopsy results only presented tissue inflammation. Thus, several further endoscopic biopsy were performed through posterior pharyngeal wall. The patient was eventually diagnosed with mucormycosis and associated osteomyelitis with subsequent bone necrosis. With confirmed diagnosis, partial maxillectomy of the necrosed bone was performed under general anesthesia. At the 4 week follow-up, the patient showed full mucosal healing and no recurrence or aggravation of the maxilla and cranial base lesion was observed.

Conclusions

Accurate diagnosis of atypical symptoms, timely diagnosis, and proper combination therapy of surgical intervention, antifungal agent, and antibiotic use for skull base osteomyelitis are all critical for proper treatment planning. In addition, biopsy and CT scans are essential in differentiating osteonecrosis from malignancy.
Literature
1.
go back to reference Chan L-L, Singh S, Jones D, Diaz EM Jr, Ginsberg LE. Imaging of mucormycosis skull base osteomyelitis. Am J Neuroradiol. 2000;21(5):828–31.PubMed Chan L-L, Singh S, Jones D, Diaz EM Jr, Ginsberg LE. Imaging of mucormycosis skull base osteomyelitis. Am J Neuroradiol. 2000;21(5):828–31.PubMed
2.
go back to reference Silverman CS, Mancuso AA. Periantral soft-tissue infiltration and its relevance to the early detection of invasive fungal sinusitis: CT and MR findings. Am J Neuroradiol. 1998;19(2):321–5.PubMed Silverman CS, Mancuso AA. Periantral soft-tissue infiltration and its relevance to the early detection of invasive fungal sinusitis: CT and MR findings. Am J Neuroradiol. 1998;19(2):321–5.PubMed
3.
go back to reference Kim J-W, Kim S-J, Kim M-R. Leucocyte-rich and platelet-rich fibrin for the treatment of bisphosphonate-related osteonecrosis of the jaw: a prospective feasibility study. Br J Oral Maxillofac Surg. 2014;52(9):854–9.CrossRef Kim J-W, Kim S-J, Kim M-R. Leucocyte-rich and platelet-rich fibrin for the treatment of bisphosphonate-related osteonecrosis of the jaw: a prospective feasibility study. Br J Oral Maxillofac Surg. 2014;52(9):854–9.CrossRef
4.
go back to reference Kim J-W, Kim S-J, Kim M-R. Simultaneous application of bone morphogenetic protein-2 and platelet-rich fibrin for the treatment of bisphosphonate-related osteonecrosis of jaw. J Oral Implantol. 2016;42(2):205–8.CrossRef Kim J-W, Kim S-J, Kim M-R. Simultaneous application of bone morphogenetic protein-2 and platelet-rich fibrin for the treatment of bisphosphonate-related osteonecrosis of jaw. J Oral Implantol. 2016;42(2):205–8.CrossRef
5.
go back to reference Park J-H, Kim J-W, Kim S-J. Does the addition of bone morphogenetic protein 2 to platelet-rich fibrin improve healing after treatment for medication-related osteonecrosis of the jaw? J oral Maxillofac Surg. 2017;75(6):1176–84.CrossRef Park J-H, Kim J-W, Kim S-J. Does the addition of bone morphogenetic protein 2 to platelet-rich fibrin improve healing after treatment for medication-related osteonecrosis of the jaw? J oral Maxillofac Surg. 2017;75(6):1176–84.CrossRef
6.
go back to reference Chandler JR, Grobman L, Quencer R, Serafini A. Osteomyelitis of the base of the skull. Laryngoscope. 1986;96(3):245–51.CrossRef Chandler JR, Grobman L, Quencer R, Serafini A. Osteomyelitis of the base of the skull. Laryngoscope. 1986;96(3):245–51.CrossRef
7.
go back to reference Grobman LR, Casiano R, Goldberg S, Ganz W. Atypical osteomyelitis of the skull base. Laryngoscope. 1989;99(7):671–6.CrossRef Grobman LR, Casiano R, Goldberg S, Ganz W. Atypical osteomyelitis of the skull base. Laryngoscope. 1989;99(7):671–6.CrossRef
8.
go back to reference Kountakis SE, Kemper JV Jr, Chang CYJ, DiMaio DJM, Stiernberg CM. Osteomyelitis of the base of the skull secondary to Aspergillus. Am J Otolaryngol. 1997;18(1):19–22.CrossRef Kountakis SE, Kemper JV Jr, Chang CYJ, DiMaio DJM, Stiernberg CM. Osteomyelitis of the base of the skull secondary to Aspergillus. Am J Otolaryngol. 1997;18(1):19–22.CrossRef
9.
go back to reference Gamba JL, Woodruff WW, Djang WT, Yeates AE. Craniofacial mucormycosis: assessment with CT. Radiology. 1986;160(1):207–12.CrossRef Gamba JL, Woodruff WW, Djang WT, Yeates AE. Craniofacial mucormycosis: assessment with CT. Radiology. 1986;160(1):207–12.CrossRef
10.
go back to reference Kohn R, Hepler R. Management of limited rhino-orbital mucormycosis without exenteration. Ophthalmology. 1985;92(10):1440–4.CrossRef Kohn R, Hepler R. Management of limited rhino-orbital mucormycosis without exenteration. Ophthalmology. 1985;92(10):1440–4.CrossRef
11.
go back to reference Yousem DM, Galetta SL, Gusnard DA, Goldberg HI. MR findings in rhinocerebral mucormycosis. J Comput Assist Tomogr. 1989;13(5):878–82.CrossRef Yousem DM, Galetta SL, Gusnard DA, Goldberg HI. MR findings in rhinocerebral mucormycosis. J Comput Assist Tomogr. 1989;13(5):878–82.CrossRef
12.
go back to reference Higgins KA, Hoang JK, Roach MC, Chino J, Yoo DS, Turkington TG, et al. Analysis of pretreatment FDG-PET SUV parameters in head-and-neck cancer: tumor SUVmean has superior prognostic value. Int J Radiat Oncol Biol Phys. 2012;82(2):548–53.CrossRef Higgins KA, Hoang JK, Roach MC, Chino J, Yoo DS, Turkington TG, et al. Analysis of pretreatment FDG-PET SUV parameters in head-and-neck cancer: tumor SUVmean has superior prognostic value. Int J Radiat Oncol Biol Phys. 2012;82(2):548–53.CrossRef
13.
go back to reference Lowe VJ, Hoffman JM, DeLong DM, Patz EF, Coleman RE. Semiquantitative and visual analysis of FDG-PET images in pulmonary abnormalities. J Nucl Med. 1994;35(11):1771–6.PubMed Lowe VJ, Hoffman JM, DeLong DM, Patz EF, Coleman RE. Semiquantitative and visual analysis of FDG-PET images in pulmonary abnormalities. J Nucl Med. 1994;35(11):1771–6.PubMed
14.
go back to reference Lowe VJ, Duhaylongsod FG, Patz EF, Delong DM, Hoffman JM, Wolfe WG, et al. Pulmonary abnormalities and PET data analysis: a retrospective study. Radiology. 1997;202(2):435–9.CrossRef Lowe VJ, Duhaylongsod FG, Patz EF, Delong DM, Hoffman JM, Wolfe WG, et al. Pulmonary abnormalities and PET data analysis: a retrospective study. Radiology. 1997;202(2):435–9.CrossRef
15.
go back to reference Lowe VJ, Fletcher JW, Gobar L, Lawson M, Kirchner P, Valk P, et al. Prospective investigation of positron emission tomography in lung nodules. J Clin Oncol. 1998;16(3):1075–84.CrossRef Lowe VJ, Fletcher JW, Gobar L, Lawson M, Kirchner P, Valk P, et al. Prospective investigation of positron emission tomography in lung nodules. J Clin Oncol. 1998;16(3):1075–84.CrossRef
Metadata
Title
Mucormycosis originated total maxillary and cranial base osteonecrosis: a possible misdiagnosis to malignancy
Authors
Young Long Park
Sura Cho
Jin-Woo Kim
Publication date
01-12-2021
Publisher
BioMed Central
Published in
BMC Oral Health / Issue 1/2021
Electronic ISSN: 1472-6831
DOI
https://doi.org/10.1186/s12903-021-01411-8

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