J Korean Assoc Oral Maxillofac Surg.  2015 Feb;41(1):48-51. 10.5125/jkaoms.2015.41.1.48.

Low-grade osteosarcoma arising from cemento-ossifying fibroma: a case report

• Lee YB
• Kim NK
• Kim JY
• Kim HJ

• KMID: 2328654
• DOI: http://doi.org/10.5125/jkaoms.2015.41.1.48

Abstract

Cemento-ossifying fibromas are benign tumors, and, although cases of an aggressive type have been reported, no cases of cemento-ossifying fibroma transforming into osteosarcoma have been documented previously. Low-grade osteosarcoma is a rare type of primary bone tumor, representing 1%-2% of all osteosarcomas. A 45-year-old female patient was diagnosed with cemento-ossifying fibroma, treated with mass excision several times over a period of two years and eight months, and followed up. After biopsy gathered because of signs of recurrence, she was diagnosed with low-grade osteosarcoma. The patient underwent wide excision, segmental mandibulectomy, and reconstruction with fibula free flap. The aim of this report is to raise awareness of the possibility that cemento-ossifying fibroma can transform into osteosarcoma and of the consequent necessity for careful diagnosis and treatment planning.

Keyword

Cemento ossifying fibroma; Low-grade osteosarcoma

MeSH Terms

Biopsy
Diagnosis
Female
Fibroma*
Fibula
Free Tissue Flaps
Humans
Mandibular Osteotomy
Middle Aged
Osteosarcoma*
Recurrence

Figure

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  Fig. 1 Preoperative panorama (A) and computed tomography (B) show buccolingual cortical expansion.

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  Fig. 2 Preoperative histological image shows woven bone and cementum-like material (H&E staining, ×40).

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  Fig. 3 Panoramic images after the first operation (A), before the second operation (B), after the second operation (C), and before the third operation (D).

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  Fig. 4 A, B. Before operation, panorama and computed tomography (CT). C, D. After the final operation, panorama and CT.

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  Fig. 5 Final histological image shows bundles of spindle cells with nuclear hyperchromasia and a variable amount of osteoid and irregular bone (H&E staining, ×40).

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  Fig. 6 Follow-up panorama (postoperative day 7 months).

Reference

1. Zupi A, Ruggiero AM, Insabato L, Senghore N, Califano L. Aggressive cemento-ossifying fibroma of the jaws. Oral Oncol. 2000; 36:129–133. PMID: 10889933.

2. Diniz AF, Filho JA, Alencar Rde C, Garcia RR, Silva MR, Ribeiro-Rotta RF, et al. Low-grade central osteosarcoma of the mandible: a case study report. Oral Surg Oral Med Oral Pathol Oral Radiol Endod. 2007; 103:246–252. PMID: 17234543.

3. Demicco EG, Deshpande V, Nielsen GP, Kattapuram SV, Rosenberg AE. Well-differentiated osteosarcoma of the jaw bones: a clinicopathologic study of 15 cases. Am J Surg Pathol. 2010; 34:1647–1655. PMID: 20975343.

4. Peravali RK, Bhat HHK, Reddy S. Maxillo-mandibular cemento-ossifying fibroma: a rare case report. J Maxillofac Oral Surg. 2013; doi: 10.1007/s12663-013-0507-6.

5. Trijolet JP, Parmentier J, Sury F, Goga D, Mejean N, Laure B. Cemento-ossifying fibroma of the mandible. Eur Ann Otorhinolaryngol Head Neck Dis. 2011; 128:30–33. PMID: 20846921.

6. Hemavathy S, Roy S. Aggressive ossifying fibroma presenting as extensive mandibular swelling: a case report. Indian J Dent. 2011; 2:107–112.

7. Toro C, Millesi W, Zerman N, Robiony M, Politi M. A case of aggressive ossifying fibroma with massive involvement of the mandible: differential diagnosis and management options. Int J Pediatr Otorhinolaryngol Extra. 2006; 1:167–172.

8. Thariat J, Julieron M, Brouchet A, Italiano A, Schouman T, Marcy PY, et al. Osteosarcomas of the mandible: are they different from other tumor sites? Crit Rev Oncol Hematol. 2012; 82:280–295. PMID: 21868246.

9. Kumar A, Varshney MK, Khan SA, Rastogi S, Safaya R. Low grade central osteosarcoma--a diagnostic dilemma. Joint Bone Spine. 2008; 75:613–615. PMID: 18457982.