Abstract
Ossifying fibromas of the head and neck region are classified as cemento-ossifying fibroma (COF) (odontogenic origin), and two types of juvenile ossifying fibromas: juvenile trabecular ossifying fibroma (JTOF), and juvenile psammomatous ossifying fibroma (JPOF). The potential for recurrence in JTOF and JPOF and the discovery of newer molecular signatures necessitates accurate histological classification. Over 12 years (2005–2017), a total of 45 patients with 51 tumours were retrieved and reviewed for clinic-pathological features from the archives of a tertiary care oncology centre. Of 45 cases, COF, JTOF and JPOF comprised 13 (28.9%), 11 (24.4%) and 18 (40%) cases respectively. Three cases were unclassifiable. M: F ratio was 1:3.3, 1.1:1, 2:1 for COF, JTOF and JPOF respectively with an age range of 6–66 years (mean: 24.6, median; 18.1 years). The most common site for COF was mandible, for JTOF was maxilla, and for JPOF was ethmoid sinus. One case of mixed JTOF and JPOF histology was seen. Aneurysmal bone cyst-like areas were seen in 26.6% of cases, most commonly in JPOF. Follow up was available in 23 cases, and ranged from 4 to 207 months. Three cases of JPOF had a recurrence and one patient with JTOF had residual disease after surgery. One case of COF demonstrated increased parathyroid hormone levels. COF, JTOF, and JPOF are clinically, radiologically and histologically distinct entities. Surgical resection is the mainstay of treatment. JPOF has a higher incidence of recurrence as compared to JTOF and COF and hence needs a more aggressive follow-up.
Similar content being viewed by others
Data Availability
On request.
References
El-Mofty SK, Nelson B, Toyosawa S. Ossifying fibroma. In: El-Naggar AK, Chan JKC, Grandis JR, Takata T, Slootweg PJ, editors. WHO classification of head and neck tumours, 4th ed. Lyon: IARC; 2017. p. 251–52.
Slootweg PJ. Juvenile trabecular ossifying fibroma: an update. Virchows Arch. 2012;461(6):699–703.
Barnes L, Eveson JW, Reichart P, Sidransky D, editors. Chapter 6, Odontogenic tumours. In: WHO classification of tumours. Pathology and genetics of head and neck tumours. Lyon: IARC Press; 2005. p. 283–327.
Urs AB, Kumar P, Arora S, Augustine J. Clinicopathologic and radiologic correlation of ossifying fibroma and juvenile ossifying fibroma—an institutional study of 22 cases. Ann Diagn Pathol. 2013;17(2):198–203.
Liu Y, Wang H, You M, Yang Z, Miao J, Shimizutani K, Koseki T. Ossifying fibromas of the jaw bone: 20 cases. Dentomaxillofacial Radiol. 2010;39(1):57–63.
Jones AV, Franklin CD. An analysis of oral and maxillofacial pathology found in children over a 30-year period. Int J Paediatr Dent. 2006;16(1):19–30.
Chang CC, Hung HY, Chang JYF, Yu CH, Wang YP, Liu BY, Chiang CP. Central ossifying fibroma: a clinicopathologic study of 28 cases. J Formos Med Assoc. 2008;107(4):288–94.
Hameed M, Horvai AE, Jordan R. Soft tissue special issue: gnathic fibro-osseous lesions and osteosarcoma. Head Neck Pathol. 2020;14(1):70–82.
El-Mofty S. Psammomatoid and trabecular juvenile ossifying fibroma of the craniofacial skeleton: two distinct clinicopathologic entities. Oral Surg Oral Med Oral Pathol Oral Radiol Endod. 2002;93:296–304.
Owosho AA, Hughes MA, Prasad JL, Potluri A, Branstetter B. Psammomatoid and trabecular juvenile ossifying fibroma: two distinct radiologic entities. Oral Surg, Oral Med Oral Pathol Oral Radiol. 2014;118(6):732–8.
Wenig BM, Vinh TN, Smirniotopoulos JG, Fowler CB, Houston GD, Heffner DK. Aggressive psammomatoid ossifying fibromas of the sinonasal region. A clinicopathologic study of a distinct group of fibro-osseous lesions. Cancer. 1995;76(7):1155–65.
Speight PM, Carlos R. Maxillofacial fibro-osseous lesions. Curr Diagn Pathol. 2006;12:1–10.
Makek M. Clinical pathology of fibro-osteo-cemental lesions of the cranio-facial skeleton and jaw bones. Basel: Karger; 1983. p. 128–227.
Johnson LC, Yousefi M, Vinh TN, Heffner DK, Hyams VJ, Hartman KS. Juvenile active ossifying fibroma: its nature, dynamic and origin. Acta Otolaryngol Suppl. 1991;488:1–40.
Toyosawa S, Yuki M, Kishino M, Ogawa Y, Ueda T, Murakami S, Konishi E, Iida S, Kogo M, Komori T, Tomita Y. Ossifying fibroma vs fibrous dysplasia of the jaw: molecular and immunological characterization. Mod Pathol. 2007;20(3):389–96.
Slootweg PJ. Maxillofacial fibro-osseous lesions: classification and differential diagnosis. Semin Diagn Pathol. 1996;13(2):104–12.
Kuta AJ, Worley CM, Kaugars GE. Central cemento-ossifying fibroma of the maxillary sinus: a review of six cases. Am J Neuroradiol. 1995;16(6):1282–6.
Prado Ribeiro AC, Carlos R, Speight PM, Hunter KD, Santos-Silva AR, de Almeida OP, Vargas PA. Peritrabecular clefting in fibrous dysplasia of the jaws: an important histopathologic feature for differentiating fibrous dysplasia from central ossifying fibroma. Oral Surg Oral Med Oral Pathol Oral Radiol. 2012;114(4):503–8.
Waldron CA. Fibro-osseous lesions of the jaws. J Oral Maxillofac Surg. 1993;51:828–35.
Brannon RB, Fowler CB. Benign fibro-osseous lesions: a review of current concepts. Adv Anat Pathol. 2001;8:126–43.
Alawi F. Benign fibro-osseous diseases of the maxillofacial bones. A review and differential diagnosis. Am J Clin Pathol. 2002;118(1):50–70.
Dujardin F, Binh MB, Bouvier C, Gomez-Brouchet A, Larousserie F, De Muret A, Louis-Brennetot C, Aurias A, Coindre JM, Guillou L, Pedeutour F. MDM2 and CDK4 immunohistochemistry is a valuable tool in the differential diagnosis of lowgrade osteosarcomas and other primary fibro-osseous lesions of the bone. Mod Pathol. 2011;24(5):624–37.
Grad-Akrish S, Rachmiel A, Ben-Izhak O. SATB2 is not a reliable diagnostic marker for distinguishing between oral osteosarcoma and fibro-osseous lesions of the jaws. Oral Surg Oral Med Oral Pathol Oral Radiol. 2021;131(5):572–81.
Swain RE, Kingdom TT, DelGaudio JM, Muller S, Grist WJ. Meningiomas of the paranasal sinuses. Am J Rhinol. 2001;15:27–30.
Pimenta FJ, Gontijo Silveira LF, Tavares GC, Silva AC, Perdigão PF, Castro WH, Gomez MV, Teh BT, De Marco L, Gomez RS. HRPT2 gene alterations in ossifying fibroma of the jaws. Oral Oncol. 2006;42(7):735–9.
de Mesquita Netto AC, Gomez RS, Diniz MG, Fonseca-Silva T, Campos K, De Marco L, Carlos R, Gomes CC. Assessing the contribution of HRPT2 to the pathogenesis of jaw fibrous dysplasia, ossifying fibroma, and osteosarcoma. Oral Surg Oral Med Oral Pathol Oral Radiol. 2013;115(3):359–67.
Tabareau-Delalande F, Collin C, Gomez-Brouchet A, Bouvier C, Decouvelaere AV, de Muret A, Pagès JC, de Pinieux G. Chromosome 12 long arm rearrangement covering MDM2 and RASAL1 is associated with aggressive craniofacial juvenile ossifying fibroma and extracranial psammomatoid fibro-osseous lesions. Mod Pathol. 2015;28(1):48–56.
Acknowledgements
We would like to extend our gratitude to Dr. Rajiv S. Desai (Professor & Head, Department of Oral & Maxillofacial Pathology, Nair Hospital Dental College, Mumbai) for his valuable suggestions to improve this manuscript.
Funding
None of the authors have any relevant financial disclosures.
Author information
Authors and Affiliations
Contributions
SRN, NM, SUR: Study Concepts; SRN, NM: Study Design; SRN, NM, SUR, MB, AP, SKA, ST: Data Acquisition.
Corresponding author
Ethics declarations
Conflict of interest
All authors declare that they have no conflicts of interest.
Ethcial Approval
This is a retrospective observational study done in accordance with the ethical standards of the institutional research committee and with the 1964 Helsinki declaration and its later amendments or comparable ethical standards. No additional procedures were performed on the participants as a part of this study. All received the standard of care for their condition and was as per the ethical standards.
Informed Consent
An informed consent has been obtained from all the study participants or their guardians (only in case of minors) for the treatment received by them, and for photographing and/or televising appropriate portions of their body and the use of this information and their histopathological material for medical, educational or scientific research (including publication) purposes after sufficient anonymization of patient particulars.
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
Nagar, S.R., Mittal, N., Rane, S.U. et al. Ossifying Fibromas of the Head and Neck Region: A Clinicopathological Study of 45 Cases. Head and Neck Pathol 16, 248–256 (2022). https://doi.org/10.1007/s12105-021-01350-4
Received:
Accepted:
Published:
Issue Date:
DOI: https://doi.org/10.1007/s12105-021-01350-4