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Bilateral cemento-ossifying fibroma of the maxillary sinus

¹ Seconda Universita' degli Studi di Napoli, Facolta' di Medicina e Chirurgica, Dipartimento "F Magrassi-A Lanzara" and ² Seconda Università degli Studi di Napoli, Facoltà di Medicina e Chirurgia Istituto di Chirurgia orale e maxillo-facciale

Correspondence: Dott. Salvatore Cappabianca, Viale Farnese, 36, 80131-Napoli, Italy

	Introduction

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A 53-year-old female patient, complaining of a slowly progressive growth of the left infraorbitary region without pain and/or tenderness. Clinical examination demonstrated a mild facial asymmetry due to a infraorbitary region swelling, which was firm and covered by normal skin. The clinical exam of the oral cavity showed an elastic consistency tumefaction located in the left upper vestibular fornix at the level of molar area and extending up to the hard palate; no signs of mucosal involvement were seen. Analogous but smaller swelling was also demonstrated in the contralateral half of the hard palate.

Ortopantomography (OPT) and CT of the facial region were performed (Figures 1 and 2).

View larger version (105K): [in this window] [in a new window]   Figure 1. Ortopantomography.

View larger version (65K): [in this window] [in a new window]   Figure 2. (a) Axial and (b) coronal CT images.

	Answer

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OPT showed partial opacification of left maxillary sinus due to a generalized inhomogeneous increased thickness of sinus floor (Figure 1).

CT images showed two different multilocular, inhomogeneously hypodense entities walled in an irregularly thick sclerotic border, located at the alveolar process of both upper jaws. The right maxillary sinus appeared completely occupied by the lesion which superior margin reached the orbit floor, lifting it; no signs of intraorbital structures involvement was demonstrable. The left smaller lesion showed a little extension into the maxillary sinus extending prevalently within its anterior wall (Figure 2).

Pathological examination revealed a composite tissue consisted in lamellar bone trabeculae, scattered throughout a fibrous stroma. Inside the fibromatous stroma fibroblast-like and osteoclastic cells were present. On haematoxylin–eosin staining concentric lines of acellular basophile material, indicative of cement, were found. A diagnosis of cemento-ossifying fibroma (COF) was made.

COF is a benign, uncommon, monostotic well-defined unilocular or multilocular fibrous-osseous tumour, arising from the periodontal ligament. COF most frequently occurs in female patients (age range 10–59 years, mean 32 years) with an incidence peak in the third and fourth decades [1].

70% of cases of COF involves the mandible but a significant percentage (22%) was found in the molar region of the maxilla, ethmoidal and orbital regions and exceptionally in petrous bone [1–3]. The COF presents a radiolucent appearance in 53%, a sclerotic radio density in 7% and mixed or mottled appearance in 40% of the cases.

There are three different patterns of radiographic borders: defined lesion without sclerotic border (40%); defined lesion with sclerotic border (45%); and lesion with ill-defined border (15%) (related to a rapid growth). In the maxillary localization, the plain sinus radiograms may show a complete opacification of sinus by a homogeneous mass laterally obscuring the contour of the maxillary bone.

CT scan shows a well-demarcated soft-tissue mass having a relatively high density (higher than muscle) inhomogeneous for contextual several areas of low density and scattered calcifications. The sinus walls may be destroyed or fragmented while the orbital floor is thinned and upwardly displaced and the medial orbital wall and frontal skull base eroded; contrast enhancement did not provide additional information [1, 4, 5].

Its occurrence in anatomical regions, not associated with periodontal membrane, is unexplained. It was supposed that pluripotential mesenchymal cells could differentiate, as does the periodontal ligament, to produce calcified material resembling bone and cementum, as well the presence of ectopic periodontal membrane has been hypothesized [3].

Despite its origin in the periodontal membrane, the factors that stimulate this structure to produce cementum in an aberrant anatomical site remain controversial. Inflammation secondary to either infections or trauma has been proposed as a causative agent [2].

Occasionally COF may grow to massive size and causes serious cosmetic and functional problems. Rare reports of lesions behaving in an aggressive fashion, diagnosed as "aggressive", "juvenile" or "active" COF, have appeared in literature [1].

No cases, to our knowledge, of multiple COF have been previously reported.

Received for publication August 6, 2001. Revision received November 12, 2001. Accepted for publication November 29, 2001.

	References

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1. Su L, Weathers DR, Waldrom CA. Distinguishing features of focal cemento-ossifying dyplasia and cemento-ossifying fibromas (II). A clinical and radiologic spectrum of 316 cases. Oral Surg Oral Med Oral Pathol Oral Radiol Endod 1997;84:540–9.[Medline]
2. Brademann G, Werner JA, Jänig U, Mehdorn HM, Rudert H. Cemento-ossifying fibroma of the petromastoid region: case report and review of the literature. J Laryngol Otol 1997;111:152–5.[Medline]
3. Fanibunda K, Reed MF. Cemento-ossifying fibroma of the mandible. Dentomaxillofacial Radiol 1997;26:246–8.[Abstract]
4. Bendet E, Bakon M, Talmi YP, Tadmor R, Kronenberg J. Juvenile cemento ossifying fibroma of the maxilla. Ann Otol Rhinol Laryngol 1997;106:75–8.[Medline]
5. Engelbrecht V, Preis S, Hassler W, Lenard HG. CT and MRI of congenital sinonasal ossifying fibroma. Neuroradiology 1999;41:526–9.[CrossRef][Medline]

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