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A 60 year old female, with no medical history of interest, who was partially edentulous, was referred to our department by her dentist after an incidental finding on a panoramic radiograph of a radiolucid lesion that was intraosseous and rounded, and that measured 3 x 2 cm approximately. Its borders were well-defined and it was situated in the right mandibular angle, by the crown of a lower impacted third molar (Fig. 1A). The lesion was not interfering with the inferior dental nerve canal, which had been displaced (Fig 1B).

The patient was clinically asymptomatic. On examination, a bulging that affected both cortical areas was detected, and mucosa of a normal appearance. The sensitivity of the inferior dental nerve had not been affected. The differential diagnosis included firstly follicular or dentigerous cyst, given its relationship with the lower third impacted molar; and in second place, keratocyst, lateral periodontal cyst, botryoid odontogenic cyst and ameloblastoma, as aggressive intraosseous lesions causing osteolysis.

Glandular odontogenic cyst: differential diagnosis and management of maxillary cyst lesions

Quiste odontogénico glandular: diagnóstico diferencial y manejo de lesiones quísticas maxilares

D. López Vaquero¹, P. Infante Cossío², M. Acosta Feria¹, J. Hernández Gutiérrez¹, A. García-Perla García², J.L. Gutiérrez Pérez²

1 Médico Residente.
2 Médico Adjunto.
3 Jefe de Servicio
Servicio de Cirugía Oral y Maxilofacial. Hospital Universitario Virgen del Rocío, Sevilla. España

Correspondence

The patient was operated on using general anesthesia. Enucleation of the lesion was carried out together with extraction of the impacted third molar and curettage of the bony bed. A clinical and radiological follow-up was carried out every three months, and no evidence of recurrence was found after 36 months (Fig. 2).

The specimen had a 2 cm diameter, it was covered by a white layer with a hard consistency (Fig. 3). The result of the histopathological examination was glandular odontogenic cyst: it had a cystic cavity that was lined by flat polystratified non-keratinized epithelium, with thicker areas that appeared to have small cysts inside. In addition, the surface of this epithelium had cuboid cells of an eosinophilic nature that were sometimes ciliated. Within these microcystic cavities there was mucous with cholesterol crystals. All this was under connective tissue that was rich in collagen, and which had no inflammatory infiltrate (Fig 4).

Discussion

Glandular odontogenic cyst (GOC) is a cystic lesion of the jaw that is vary rare (0.012%) according to Magnusson and cols,¹ appearing within a wide age range (approximate mean age of 50); it has no clear sex predilection, it is more commonly found in the mandible (85%) and in the anterior sector as a radiolucid lesion. It is uni- or multilocular, with well-defined scalloped borders. It was first described by Gardner and cols² in 1988, although Padayachee and Van Wyk had described two cases a year previously with the same characteristics, which they called sialo-odontogenic cyst.³ It was Gardner and cols. who first used the name of glandular odontogenic cyst,² with which it appears in the WHO’s classification of cystic lesions of the jaw because of its probable dental origin (1992). Only some 60 cases have been described in the literature, perhaps because this entity has been recognized as such not very long ago, given that these cysts were diagnosed as different types of odontogenic tumors because of their histological, clinical and radiological similarity.⁴

The clinical case presented in this article is similar to all those that have previously been described in the literature: mandibular cystic lesion, unilocular radiolucid image, with welldefined borders. It is unusual in that it is associated with an impacted third molar, which led to the preoperative presumed diagnosis of a follicular cyst. To our knowledge it is the third case presented in the literature with this differential characteristic⁵. There are no clinical or radiological signs to suspect it, and it usually presents mimicking other entities, such as in this case of ours.

Histologically it appears as a cystic cavity that is lined by flat stratified non-keratinized epithelium, with thicker areas that appear to have small cysts inside. These also appear in the periodontal lateral cyst and in the odontogenic botryoid cyst,^6,7 which would justify the hypothesis of its possible odontogenic origin, as proposed by Gardner and cols.² This epithelium also has eosinophilic cuboid cells in its more superficial layers, which may be ciliated and form pseudopapillae. Within the microcystic cavities there are mucous pools (mucicarmine and PAS +) with a glandular appearance. This is what led Padayachee and Van Wyk to call it sialo-odontogenic cyst.³ All this is supported by connective tissue that is rich in collagen but there is no inflammatory infiltrate. These histological characteristics are similar to some entities with an odontogenic origin such as: lateral periodontal cyst, odontogenic botryoid cyst or dentigerous cyst.⁸ However, what differentiates it from the others, is the high rate of recurrence, and on occasions it has shown aggressive behavior.

The lateral periodontal cyst is a radiolucid lesion with well-defined borders, that is uni- or multilocular, adjacent to the apexes of vital teeth, and lined with a fine layer of flat stratified non-keratinized epithelium. At certain points it becomes thicker with eosinophilic glycogen-rich cells. What differentiates it from GOC is its high recurrence rate.^4,8

The botryoid odontogenic cyst has a similar histologic structure, but it has a greater tendency to recurrence, and it is therefore more aggressive. It is therefore thought that GOC is a variation of the botryoid cyst; and it is even thought that the three entities are three variants of a group of non-keratinized epithelial cysts with an odontogenic origin.⁹ The existence of ciliated epithelium and acinic ductal structures in localized areas means that, within these entities, GOC has its own name.

A similar characteristic between central mucoepidermoid carcinoma (CMC) and GOC is the intraosseous development of glandular structures. Around 30% of odontogenic cysts have mucosal cells in their interior.⁴ It is thought that CMC arises from the epithelium that covers odontogenic tumors after dysplastic phenomena of mucosal cells, dentigerous cysts in particular.⁴ Many of these tumors have a pericoronal location, simulating lesions that are more common in this area, and they appear in association with an impacted tooth, as in this case of ours. According to various reviews of the literature, the similarity between CMC and GOC has been demonstrated, with a 25% recurrence occurring, depending on the treatment carried out. Some authors have reached the conclusion that GOC could be a low grade CMC, in which the areas of epithelial thickening and microcyst formation, undergo dysplastic changes, and that they are entities that clinically and morphologically overlap.⁴

Kaplan and cols¹⁰ carried out a revision of all the treatment carried out in GOC cases that were found between 1987 and 2003. They studied location, locularity, extension, cortical integrity, treatment, follow-up time and recurrence: in 48 of the cases minor surgery was carried out (enucleation, curettage or marsupialization), and 8 major surgeries (peripheral ostectomy or en bloc resection). The follow-up that was carried out was between 3 months and 20 years, with a mean of 3 years. There was recurrence in 13 cases (27.1%). This recurrence was associated with multilocularity, large lesions and cortical bone compromise. All the patients that experienced recurrence received conservative treatment with minor surgery. None of these were patients that had initially undergone aggressive surgery.

The rate of recurrence is much greater than that of odontogenic, inflammatory or developmental cysts, and it is only similar to that of the keratocyst. Some keratocyst follow-up series indicate a recurrence of nearly 20%, which is only surpassed by the ameloblastoma. The reason that can explain this is the extreme fineness of the epithelium that lines the cystic cavity, and the presence of microcysts, making total enucleation very difficult.⁹ Various indicative factors of aggressiveness of GOC have been established10: high rate of recurrence, cortical integrity (39.3% perforation and 14.3% erosion) and multilocularity. Cortical bone compromise is associated with a high probability of recurrence. This finding makes carrying out aggressive treatment necessary in order to avoid recurrence. Chavez and Richter¹¹ studied multilocularity, finding a link with disease recurrence, suggesting that it should be converted into another parameter when taking a therapeutic decision. It has also been demonstrated that recurrence is related to the treatment carried out.¹² Kaplan and cols. demonstrated 25% recurrence with regard to enucleation and simple curettage of the lesion.¹⁰ Based on all these findings the following procedure protocol has been proposed:^10,12

• For small lesions, that are clinically and radiologically unspecific, and unilocular: enucleation and curettage with later analysis. If the lesion is totally enucleated no further surgery should be indicated. However, a follow up of a minimum of 3 years is recommended.

• Large or multilocular lesion: a biopsy should be carried out in order to make a therapeutic decision. For large but unilocular lesions, enucleation is recommended with preservation of vital structures. Marsupialization can be carried out, and also peripheral ostectomies for ensuring there is no recurrence. More aggressive treatment is recommended for larger and multilocular lesions: marginal or segmental mandibulectomy with use of bone grafts for reconstruction. If the lesion is near the maxillary sinus or nasal fossa, marsupialization is indicated. A follow-up of at least three years is recommended.

Our case is in line with the treatment that should be carried out in the first point of the protocol: small, unilocular lesion, with well-defined borders that are asymptomatic; we carried out enucleation and curettage of the cavity, not finding any evidence of recurrence after a follow-up of three years.

Conclusions

GOC is a rare entity, which histologically and radiographically is similar to other odontogenic cysts, but it has a greater rate of recurrence and aggressive behavior. It should be included in the differential diagnosis of maxillary cysts, in order for adequate treatment to be carried out and for recurrence to be avoided. A long-term follow up is compulsory.

Correspondence:
Pedro Infante Cossío
Servicio de Cirugía Oral y Maxilofacial
Hospital Universitario Virgen del Rocío
Avda. Manuel Siurot s/n
41013 Sevilla. España
E-mail: pinfante@us.es

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