Skip to main content
Top

Open Access 09-06-2024 | Osteosarcoma | Case Report

Osteosarcoma associated with cemento-osseous dysplasia: co-incidence or two related entities?

Authors: Suvarna Indermun, Fadi Titinchi, Julandi Alwan, Jean Morkel, Christoffel Johannes Nortje

Published in: Oral Radiology

Login to get access

Abstract

Background

Osteosarcoma of the jaws is a rare primary malignant tumor of bone. The clinical, radiological and histopathological features of a case associated with cemento-osseous dysplasia is presented.

Case Report

A 57-year-old mixed-race female presented with a large, progressive, swelling of the right mandible. Radiographic examination revealed two associated lesions. Partially defined irregular radiopacities were noted in the left mandible, extending from the premolar to the molar region. The lesion had a cotton-wool appearance and resembled a fibro-osseous lesion; i.e. cemento-osseous dysplasia. A second large, expansive and irregular, radiopaque lesion was noted on the right angle of the mandible, extending beyond the inferior cortex of the mandible. The internal structure was heterogeneous and resembled irregular bone formation. The classic “sunburst” appearance of radiating bony spicules can be seen in the posterior–anterior view and the CBCT 3D reconstruction, indicating the outgrowth of the tumor matrix. Histopathological exam confirmed a final diagnosis of osteosarcoma closely associated with cemento-osseous dysplasia. The patient underwent a fludeoxyglucose-18 (FDG) positron emission tomography (PET) scan which indicated metastasis in the left lung and increased uptake in the right mandible. Chemotherapy was initially administered with a plan to resect the tumor, however, the patient demised as a result of medical complications.

Conclusion

The question in the literature remains whether these two entities are coincidentally found or arise from each other. Nevertheless, it is important for clinicians to closely monitor patients with cemento-osseous dysplasia and biopsy any suspicious lesions that may develop into osteosarcoma.
Literature
3.
go back to reference Fenerty S, Shaw W, Verma R, Syed AB, Kuklani R, Yang J, et al. Florid cemento-osseous dysplasia: review of an uncommon fibro-osseous lesion of the jaw with important clinical implications. Skeletal Radiol. 2017;46(5):581–90.CrossRefPubMed Fenerty S, Shaw W, Verma R, Syed AB, Kuklani R, Yang J, et al. Florid cemento-osseous dysplasia: review of an uncommon fibro-osseous lesion of the jaw with important clinical implications. Skeletal Radiol. 2017;46(5):581–90.CrossRefPubMed
4.
go back to reference Langlais RP, Langland OE, Nortje CJ. Diagnostic imaging of the jaws. 1st ed. Malvern: Williams & Wilkins; 1995. Langlais RP, Langland OE, Nortje CJ. Diagnostic imaging of the jaws. 1st ed. Malvern: Williams & Wilkins; 1995.
5.
go back to reference Farman AG, Nortje CJ, Wood RE. Fibro-osseous Lesions. In: Oral and maxillofacial diagnostic imaging. 1st ed. St Louis: Mosby; 1993. p. 319. Farman AG, Nortje CJ, Wood RE. Fibro-osseous Lesions. In: Oral and maxillofacial diagnostic imaging. 1st ed. St Louis: Mosby; 1993. p. 319.
8.
go back to reference Li B-B, Zhang J-Y, Gao Y. Chondroblastic osteosarcoma arising in the maxilla mimicking the radiographic and histological characteristics of cemento-osseous lesions: a case report. Mol Clin Oncol. 2017;6(5):733–6.CrossRefPubMedPubMedCentral Li B-B, Zhang J-Y, Gao Y. Chondroblastic osteosarcoma arising in the maxilla mimicking the radiographic and histological characteristics of cemento-osseous lesions: a case report. Mol Clin Oncol. 2017;6(5):733–6.CrossRefPubMedPubMedCentral
9.
go back to reference Hameed M, Horvai AE, Jordan RCK. Soft tissue special issue: gnathic fibro-osseous lesions and osteosarcoma. Head Neck Pathol [Internet]. 2020;14:70–82.CrossRefPubMed Hameed M, Horvai AE, Jordan RCK. Soft tissue special issue: gnathic fibro-osseous lesions and osteosarcoma. Head Neck Pathol [Internet]. 2020;14:70–82.CrossRefPubMed
10.
go back to reference Savage SA, Mirabello L. Using epidemiology and genomics to understand osteosarcoma etiology. Sarcoma. 2011;2011:14–6.CrossRef Savage SA, Mirabello L. Using epidemiology and genomics to understand osteosarcoma etiology. Sarcoma. 2011;2011:14–6.CrossRef
11.
go back to reference Wagner VP, Carlos R, Romañach MJ, Lopes MA, Speight PM, Vargas PA. Malignant transformation of craniomaxillofacial fibro-osseous lesions: a systematic review. J Oral Pathol Med. 2019;48(6):441–50.CrossRefPubMed Wagner VP, Carlos R, Romañach MJ, Lopes MA, Speight PM, Vargas PA. Malignant transformation of craniomaxillofacial fibro-osseous lesions: a systematic review. J Oral Pathol Med. 2019;48(6):441–50.CrossRefPubMed
12.
go back to reference Lam EWN. Malignant neoplasms. In: Mallya SM, Lam EWN, editors. White & pharoah’s oral radiology: principles and interpretation. 8th ed. St Louis: Elsevier; 2019. p. 493–504. Lam EWN. Malignant neoplasms. In: Mallya SM, Lam EWN, editors. White & pharoah’s oral radiology: principles and interpretation. 8th ed. St Louis: Elsevier; 2019. p. 493–504.
13.
go back to reference Lam EWN. Diseases affecting the structure of bone. In: Mallya SM, Lam EWN, editors. White & pharoah’s oral radiology: principles and interpretation. 8th ed. St Louis: Elsevier; 2019. p. 475–85. Lam EWN. Diseases affecting the structure of bone. In: Mallya SM, Lam EWN, editors. White & pharoah’s oral radiology: principles and interpretation. 8th ed. St Louis: Elsevier; 2019. p. 475–85.
14.
go back to reference Singer SR, Mupparapu M, Rinaggio J. Florid cementoosseous dysplasia and chronic diffuse osteomyelitis report of a simultaneous presentation and review of the literature. J Am Dent Assoc Internet. 2005;136(7):927–31.CrossRef Singer SR, Mupparapu M, Rinaggio J. Florid cementoosseous dysplasia and chronic diffuse osteomyelitis report of a simultaneous presentation and review of the literature. J Am Dent Assoc Internet. 2005;136(7):927–31.CrossRef
15.
go back to reference Schneider LC, Dolinsky HB, Grodjesk JE, Mesa ML, Doyle JL. Malignant spindle cell tumor arising in the mandible of a patient with florid osseous dysplasia. Oral Surg Oral Med Oral Pathol Oral Radiol Endod. 1999;88(1):69–73.CrossRefPubMed Schneider LC, Dolinsky HB, Grodjesk JE, Mesa ML, Doyle JL. Malignant spindle cell tumor arising in the mandible of a patient with florid osseous dysplasia. Oral Surg Oral Med Oral Pathol Oral Radiol Endod. 1999;88(1):69–73.CrossRefPubMed
16.
go back to reference Melrose R, Handlers J. Osteosarcoma ex florid osseous dysplasia: report of a case. Oral Surgery, Oral Med Oral Pathol Oral Radiol Endodontology. 2003;96(September):296.CrossRef Melrose R, Handlers J. Osteosarcoma ex florid osseous dysplasia: report of a case. Oral Surgery, Oral Med Oral Pathol Oral Radiol Endodontology. 2003;96(September):296.CrossRef
17.
18.
go back to reference Lee RJ, Arshi A, Schwartz HC, Christensen RE. Characteristics and prognostic factors of osteosarcoma of the jaws a retrospective cohort study. JAMA Otolaryngol—Head Neck Surg. 2015;141(5):470–7.CrossRefPubMed Lee RJ, Arshi A, Schwartz HC, Christensen RE. Characteristics and prognostic factors of osteosarcoma of the jaws a retrospective cohort study. JAMA Otolaryngol—Head Neck Surg. 2015;141(5):470–7.CrossRefPubMed
19.
go back to reference Padilla RJ, Murrah VA. The spectrum of gnathic osteosarcoma: caveats for the clinician and the pathologist. Head Neck Pathol. 2011;5(1):92–9.CrossRefPubMed Padilla RJ, Murrah VA. The spectrum of gnathic osteosarcoma: caveats for the clinician and the pathologist. Head Neck Pathol. 2011;5(1):92–9.CrossRefPubMed
20.
go back to reference Reith JD. Bone and Joints. In: Goldblum JR, Lamps LW, McKenney JK, Myers JL, editors. Rosai and ackerman’s surgical pathology. 11th ed. New York: Elsevier; 2003. p. 1752–9. Reith JD. Bone and Joints. In: Goldblum JR, Lamps LW, McKenney JK, Myers JL, editors. Rosai and ackerman’s surgical pathology. 11th ed. New York: Elsevier; 2003. p. 1752–9.
21.
go back to reference Lopes M, Baumhoer D, Jay A. Osteosarcoma of the jaw. In: WHO classification of head and neck tumours. 5th ed. Lyon: International Agency for Research on Cancer; 2022. p. 224. Lopes M, Baumhoer D, Jay A. Osteosarcoma of the jaw. In: WHO classification of head and neck tumours. 5th ed. Lyon: International Agency for Research on Cancer; 2022. p. 224.
Metadata
Title
Osteosarcoma associated with cemento-osseous dysplasia: co-incidence or two related entities?
Authors
Suvarna Indermun
Fadi Titinchi
Julandi Alwan
Jean Morkel
Christoffel Johannes Nortje
Publication date
09-06-2024
Publisher
Springer Nature Singapore
Published in
Oral Radiology
Print ISSN: 0911-6028
Electronic ISSN: 1613-9674
DOI
https://doi.org/10.1007/s11282-024-00758-2