Glandular odontogenic cyst (GOC) is an uncommon developmental cyst of the jaw with only 200 cases have been reported so far.1 This case report presented a GOC case in the left premolar region of the mandible.
A 84-year-old male patient was referred for evaluation of a unilocular radiolucent lesion at the left mandible. The patient was asymptomatic and the lesion was found incidentally in a panoramic radiograph (Fig. 1A). His past medical history included sebaceous carcinoma at the left eyelid awaiting for radiation therapy. Oral examination showed teeth 33 and 34 retained root, teeth 32 and 35 periodontitis affected teeth. No swelling was observed. Additional computerized tomography (CT) scan revealed a well-defined radiolucent lesion in the left body of mandible, extending from teeth 32 to 35 with slight buccolingual cortical plate expansion and focal perforation of buccal cortex (Fig. 1B). Radicular cyst, odontogenic keratocyst, and ameloblastoma were included in the clinical differential diagnosis of the lesion. After discussing the treatment plan with the patient and obtaining the signed informed consent, the lesion was completely excised under local anesthesia (Fig. 1C). The excised specimen was sent for histopathological examination. Microscopically, the specimen showed fragments of fibrocollagenous vascular connective tissue partially lined by non-keratinized stratified squamous epithelium. Moreover, ciliated cuboidal epithelial cells and mucous cells were observed throughout the lesion (Fig. 1D). The epithelium exhibited focal areas of nodular thickening (Fig. 1E, F, and G) and small intraepithelial microcystic spaces (Fig. 1H). The characteristic histopathological features confirmed the final diagnosis of a glandular odontogenic cyst.
Figure 1.
The radiographic images, excised specimen, and histopathological microphotographs of the present case of glandular odontogenic cyst. (A) Panoramic radiograph revealed a well-defined unilocular radiolucent lesion extending from teeth 32 to 35. (B) CT image showed a unilocular radiolucent lesion with slight buccolingual cortical plate expansion and focal perforation of buccal cortex. (C) The excised specimen showed an irregular-shaped cystic lesion. (D) The high-power image showed fragments of fibrous connective tissue partially lined by non-keratinized stratified squamous epithelium. The mucous cells were observed throughout the epithelium (red arrow). (E and F) The medium and high-power images showed the epithelium exhibited focal areas of nodular thickening into the lumen (green arrow). (G) Another area of epithelial nodular thickening was observed. (H) Small intraepithelial microcystic spaces were observed in the epithelial lining (blue arrow). Original magnification for figure E 100 × image and for figures D, F, G, and H 400 × images. (For interpretation of the references to colour in this figure legend, the reader is referred to the Web version of this article.)
Glandular odontogenic cyst (GOC) is a rare developmental cyst of the jaws, accounting for <0.5% of all odontogenic cysts.1,2 Clinically, the most common site of occurrence is the anterior mandible. The GOC occurs mostly in the 5th – 7th decade of life.2 There is no sex predilection.2 The most common presentation is an asymptomatic, slow growing mass of the jaws.1,2 Radiographically, the lesion may appear as a well-defined unilocular or multilocular radiolucent lesion, with or without scalloped border.3 Cortical expansion and root resorption may present.2,3 The lesion may cross the midline in the mandible.2,3 The histopathological features for the GOC were variable thickness and types of lining epithelium.1,2,4 The epithelial lining could be simple squamous or cuboidal cells to stratified squamous epithelium with focal thickening in the cystic lining.1,2,4 Intraepithelial microcysts and mucous cells are frequently observed.1,2,4 Importantly, GOC may show some histopathologic features similar to central mucoepidermoid carcinoma (CMEC).4 However, GOC shows the cystic spaces lined by epithelium of variable thickness without any solid epidermoid component as observed in CMEC. In addition, CMEC does not show plaque-like epithelial thickening which is usually seen in GOC.4 Moreover, GOC has been found to be negative for MAML2 gene rearrangement.4 The molecular pathogenesis of GOC remains unclear and needs to be further elucidated.4,5 Enucleation is a common treatment for GOC. However, due to high recurrence rate of GOC, the long-term follow-up or using aggressive treatment modalities are recommended.4
Declaration of competing interest
The authors have no conflicts of interest relevant to this article.
Acknowledgments
None.
References
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