Quiste odontogénico calcificante asociado con odontoma complejo: Presentación de un caso y revisión bibliográfica

Gallana Alvarez, Silvia; Mayorga Jimenez, Francisco; Torres Gómez, Francisco Javier; Avellá Vecino, Francisco Javier; Salazar Fernandez, Clara

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Medicina Oral, Patología Oral y Cirugía Bucal (Ed. impresa)

versión impresa ISSN 1698-4447

Med. oral patol. oral cir. bucal (Ed.impr.) vol.10 no.3 may./jul. 2005

Calcifying odontogenic cyst associated with complex odontoma:
Case report and review of the literature
Quiste odontogénico calcificante asociado con odontoma complejo: Presentación de un caso y revisión bibliográfica

Silvia Gallana Alvarez ⁽¹⁾, Francisco Mayorga Jimenez ⁽²⁾, Francisco Javier Torres Gómez ⁽³⁾, Francisco Javier Avellá Vecino ⁽⁴⁾,
Clara Salazar Fernandez ⁽⁵⁾

(1) Médico Interno Residente (3ª año). Servicio de Cirugía Maxilofacial
(2) Médico Adjunto. Servicio de Cirugía maxilofacial
(3) Médico Interno Residente (3º año). Servicio de Anatomia Patológica
(4) Médico Interno Residente (5ª año). Servicio de Cirugía Maxilofacial
(5) Médico Adjunto. Servicio de Cirugía maxilofacial. Hospital Universitario Virgen Macarena. Sevilla

Address:
Silvia Gallana Alvarez
C/ Flor de papel B 5 P 13 1ªB
41020 SEVILLA.
Tfno: 955008963 / 678525000
Fax: 955008949
E-mail: sgallana@hotmail.com.

Received: 11-12-2003 Accepted: 4-04-2004

Gallana-Alvarez S, Mayorga-Jimenez F, Torres-Gómez FJ, Avellá-Vecino FJ, Salazar-Fernandez C. Calcifying odontogenic cyst associated with complex odontoma: Case report and review of the literature. Med Oral Patol Oral Cir Bucal 2005;10:243-7.
© Medicina Oral S. L. C.I.F. B 96689336 - ISSN 1698-4447

SUMMARY
We report a calcifying odontogenic cyst associated with odontoma (COCaO) and an included permanent canine in the superior maxilla, in a 19 year-old-man.
The calcifying odontogenic cyst (COC) was first described as a distinct entity by Gorlin et al in 1962.
The lesion is a mixed odontogenic benign tumor, and although most of the cases present cystic characteristics, a few are of the solid type (15%), and its rare malignant transformation is well documented.
The COC may occur in association with other odontogenic tumors, the most common is the odontoma, occurring in about 24% of the cases. For this association the term Odontocalcifying odontogenic cyst has been suggested.
Radiographically is a well defined mixed lesion and histologically consists of a large cyst. In the central area of the cyst enamel and dentin deposits can be found, irregularly distributed in areas and in other parts it takes on a well defined organoid aspect.
A thorough review of literature takes place and the pathogenesis is discussed.

Key words: Calcifying odontogenic cyst, Gorlin´s cyst, odontogenic cysts, odontogenic tumors.

RESUMEN

Se presenta un caso de quiste odontogénico calcificante asociado con odontoma (QOCaO) y con un canino permanente incluido en maxilar superior, en un paciente varón de 19 años.
El Quiste Odontogénico Calcificante (QOC) fue descrito por primera vez cómo una entidad patológica por Gorlin y col. en 1962.
Se trata de un tumor benigno odontogénico mixto,y aunque la mayoria de estas lesiones presentan características quísticas en algunos casos se presenta como una masa sólida (15 %), y su transformación maligna está bien documentada. El QOC se presenta en ocasiones asociado a otros tumores odontogénicos, el más común es el odontoma al que aparece relacionado en el 24% de los casos. Para esta asociación se ha sugerido el término de "Quiste Odontogénico Odontocalcificante".
Radiológicamente es una lesión mixta, bien definida y que histológicamente consiste en un gran quiste en cuya parte central existe un área sólida con acúmulos de esmalte y dentina distribuidos irregularmente en algunas zonas y en otros lugares adopta un aspecto organoide bien definido.
Se efectúa una revisión de la literatura y se discute su patogénesis.

Palabras clave: Quiste odontogénico calcificante, quiste de Gorlin, quistes odontogénicos, tumores odontogénicos.

INTRODUCTION

The COC was first described as a distinct entity by Gorlin et al in 1962 (1).

This lesion is uncommon and represents about 0.03% of the biopsy lesions and less than 2% of all odontogenic cysts and tumors(2,3). A great variety of clinical and histologic features have been reported and several classifications have been proposed (4,5). Most cases of COC have features of a cyst but in about 15% of the cases they are solid lesions (3,6).

In 1992 the World Health Organization (WHO) classified COC within the groups of neoplasm and tumors that originate from odontogenic tissues (7,8), but confirmed that most of the cases are non-neoplastic.

The COC normally appears as a painless, slow-growing tumor, affecting equally the maxilla and mandible, with predilection to the anterior segment (incisor/canine area). It generally affects young adults in the third to fourth decade, without gender predilection (8,9).

Radiographically, COC is usually a mixed lesion, with radiolucent area, uni or multilocular, that contains different amounts of radiopaque material. Association with impacted teeth is described in 10-32% (4,10) of the cases. Radicular reabsorption is not common (10,11).

Histologically, COC is usually composed of a cystic cavity with a fibrous capsule, lined by an odontogenic epithelium (12). The typical microscopic characteristic of this lesion is the presence of variable amounts of aberrant epithelial cells, without nuclei, which are named "ghost cells". In addition displastic dentine can be found and occasionally the cyst is associated with an area of dental hard tissue formation resembling an odontoma (7).

The COC-Odontoma association has been reported in 24% of the cases. This lesion can also be found associated with other odontogenic tumors, like ameloblastomas, ameloblastic fibroodontoma, odontoameloblastic tumor, calcifying epithelial odontogenic tumor and adenomatoid odontogenic tumor (13,14).

CASE REPORT

We report the case of a 19-year-old man, without any remarkable medical history, who was refered by his dentistry to our clinic because he still had temporary canine without any other accompanying symptoms.

By the intraoral inspection a light tumefaction was observed at the canine fosa, as well as the absence of the definitive canine. The mucosa aspect was absolutely normal.

The definitive included canine and a well defined unilocular radiolucent area extending from medial line to the premolar region, with radiopaque mass, was observed in the OPG and in the lateral craneal radiography (Fig1). With these findings surgical resection was recommended.

A thick wall cyst was completely removed under local anaesthetic, with a dentin magma compatible with a complex odontoma inside the cyst and an included permanent canine.

The pathologic report showed a cystic lesion with 2 cm of highest diameter, soft consistence and browndish colouring.

Microscopic examination showed a cystic cavity lined by a prominent and well defined basal layer of cuboidal and columnar cells that focally resemble ameloblastic ephithelium. Overlying this layer we can find epithelial cells similar to the central stellate reticulum of the tooth germen and between them, the typical "Ghost cell" (large eosinophilic cells with aberrant keratinisation) (Fig 2). Foreign body gigant cells are found in close relationship to the aberrant keratin.

There is a complex odontoma related to the calcifying odontogenic cyst, that is comprised of a mixture of dental hard tissues with no resemblance to a tooth, dentina the predominant tissue, enamel matrix, cementun and connective tissue (Fig 3).

The histopathologic diagnosis was calcifying odontogenic cyst associated with complex odontoma.

We reported a normal clinical healing after surgical treatment and 28 months of follow-up, recurrence was not found at the moment.

DISCUSSION

The review of literature shows that COCs are rare lesions that affect men and women equally, and it appears most frequently in the mandible (15).

COC may occur in association with other odontogenic tumors, the most common of these is the odontoma. Buchner (4) shows this association in 35% of his cases, Nagao et al in 22% (16) and Shamaskin et al in 47% (17).

Hirshberg (15) reveals 52 cases of COC associated with odontoma, analyzes clinical and histological features, and it was classifies as a separate entity. The term "odontocalcifying odontogenic cyst" was suggested. The anterior region of the jawbones was the most commonly affected site in both cases, but the COCaO presents a female predominance (2:1) with a mean age of 16 years, most frequently occuring in the maxilla (61´5%).

Radiographically appears as a mixed radiolucent-radiopaque lesion (80´5%). Microscopically the epithelial components in COCaO were identical to those described for simple COC, but the former presents tooth-like structures that appear to be an integral part of the lesion.

Several possibilities are suggested regarding the pathogenesis of COCaO. One possibility is that the COC and the odontoma may represent coincidental juxtaposition of COC and an odontoma, because other odontogenic tumors like ameloblastoma have been reported to be associated with COC (17,18).

Other investigators suggest that the COC develops secondarily from odontogenic epithelium that participates in the formation of the odontoma (14,16,19). However, it also has been suggested that the odontoma develops secondarily from lining epithelium of the COC (3,20).

And finally some authors think that the COCaO should be regarded as a separate entity (15), characterized by the clinical differences in location and in age distribution between simple COC and COCaO.

The cytokeratin expression in the COCaO has been studied by several authors (8,21), confirming its odontogenic origin.

The COCaO should be treated conservatively by surgical enucleation because recurrences are very uncommon(9,22-24).

In our case, our treatment involved surgical conservative treatment and a follow-up for 28 months. We observed normal healing and absence of recurrence.

Finally, the malignant transformation of a preexisting benign COC could happen, but is extremely uncommon (25,26).

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