| Abstract|| |
Glandular odontogenic cyst (GOC) is a recently recognized rare developmental odontogenic cyst having an aggressive behavior and accounting for 0.012% to 1.3% of all jaw cysts. GOC usually presents as a painless, slow-growing swelling that tends to affect the anterior part of the jaws. It chiefly occurs in the fourth and fifth decades of life and presents as an expansion of jaws with or without pain or paresthesia. Aggressive nature of the lesion has been reported, as supported by the fact that 25 to 55% of cases recur following curettage. So far only just over 113 cases of GOC have been reported in the literature. Here, we report a case of bilateral GOC in the posterior region of the maxilla, in a 29-year-old male patient, which is unique, being the first case of bilateral GOC to be reported in the literature.
Keywords: Bilateral, glandular odontogenic cyst, maxilla
|How to cite this article:|
Amberkar VS, Jahagirdar A, Ahmed Mujib B R. Glandular odontogenic cyst: Report of an unusual bilateral occurrence. Indian J Dent Res 2011;22:364
|How to cite this URL:|
Amberkar VS, Jahagirdar A, Ahmed Mujib B R. Glandular odontogenic cyst: Report of an unusual bilateral occurrence. Indian J Dent Res [serial online] 2011 [cited 2019 Oct 22];22:364. Available from: http://www.ijdr.in/text.asp?2011/22/2/364/84285
| Case Report|| |
A 29-year-old man reported to our hospital with complaint of pain in the upper right back region of the jaw for a period of 4 to 5 days. The patient was asymptomatic earlier, and the pain had aggravated on chewing hard food. Extraorally, the swelling was not obvious and did not produce any facial asymmetry. Intraorally, the swelling was firm in consistency, extending from distal aspect of canine to second molar on both the sides. Mucosa over the swelling appeared normal. Tenderness on percussion was elicited in teeth 14 and 15. Radiography revealed periapical radiolucency in relation to 14, 15, 16; and 24, 25, 26; measuring approximately 5×6 cm in size [Figure 1]. Intraoral periapical radiograph (IOPA) of the left side lesion showed divergence of root in 14 and 15[Figure 2]. An excisional biopsy of both the lesions was performed, and the specimen was submitted for histopathological examination.
|Figure 1: Occlusal radiograph showing unilocular radiolucency with well-defined borders|
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Macroscopically, the submitted tissues were slimy in consistency and whitish gray in color. Microscopically, both the specimens showed similar histological features and revealed a non-keratinized stratified squamous epithelial cystic lining surrounded by a connective tissue capsule. The cystic lining was of variable thickness, exhibiting focal plaques of epithelium with a flat connective tissue interface. The superficial layer of the cystic lining consisted of columnar cells, goblet cells and a few ciliated cells that formed papillary projections. Within the thickness of the epithelium, areas exhibiting intra-epithelial gland-like structures were observed [Figure 3]. Connective tissue revealed the formation of daughter cysts[Figure 4]. Few areas of irregular calcifications and mild chronic inflammatory cell infiltration were also observed.
|Figure 3: Right maxillary lesion showing epithelial plaques and intra-epithelial gland-like structures [H and E, ×40]|
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|Figure 4: Left maxillary lesion showing superficial layer of cystic lining consisting of columnar cells, goblet cell with a few ciliated cells and daughter cysts (H and E, ×40)|
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The presence of mucin within the intra-epithelial gland-like structures was confirmed by mucicarmine staining [Figure 5]. Mucin was further characterized using periodic acid-Schiff (PAS) stain, and mucous pooling showed positivity to PAS, suggestive of acidic mucin [Figure 6]. The final diagnosis of glandular odontogenic cyst was arrived by correlating with the clinical, radiographic, and histopathologic features.
|Figure 5: Intra-epithelial gland-like structures showing positivity for mucicarmine stain, ×10|
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|Figure 6: Mucous pooling demonstrated by periodic acid-Schiff stain, ×10|
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| Discussion|| |
GOC is a recently recognized rare developmental odontogenic cyst having an aggressive behavior and accounting for 0.012% to 1.3% of all jaw cysts.  GOC usually presents as a painless, slow-growing swelling that tends to affect the anterior part of the jaws.  Aggressive nature of the lesion has been reported, as supported by the fact that 25% to 55% of cases recur following curettage. 
Padayachee and Van Wyk, in 1987 coined the term sialo-odontogenic cyst for GOC, as histologic features of these cysts were suggestive of glandular differentiation along with features of the botryoid variant of lateral periodontal cyst.  In 1988, Gardner et al. reported a series of these cysts and called the lesion glandular odontogenic cyst because the cyst wall epithelium contained only mucin elements with no evidence of salivary tissue involvement.  In 1992, GOC was included in the WHO histologic typing of odontogenic tumors under the term glandular odontogenic cyst or sialo-odontogenic cyst and was recognized by the WHO as "a cyst arising in the tooth-bearing areas of the jaws characterised by an epithelial lining with cuboidal or columnar cells both at the surface and lining crypts or cyst-like spaces within the thickness of the epithelium" 
The predominance of mandibular involvement in GOC has been reported previously with predilection for the anterior areas of the jaws.  Bilateral occurrence of GOC has not been reported in the literature till now. In the present case, the lesions were bilateral and present in the posterior maxilla.
The present case fulfils the microscopic criteria for the diagnosis of GOC, given by Gardner et al.  such as presence of cystic lining with cuboidal cells with papillary luminal proliferation as well as presence of characteristic epithelial plaque and mucicarmine positive material in the intra-epithelial crypts.
GOC should be distinguished from lateral periodontal cyst, botryoid odontogenic cyst, and central mucoepidermoid carcinoma (multicystic variant) as it exhibits considerable overlapping of histological features.  Presence of ciliated epithelium and duct-like spaces with mucous cells specifically differentiates GOC from lateral periodontal cyst and botryoid odontogenic cyst. To differentiate low-grade central mucoepidermoid carcinoma from GOC is more important and can be difficult. Presence of typical thin lining of epithelium without any solid epithelial proliferation and the evidence of epithelial plaques differentiate GOC from central mucoepidermoid carcinoma.
Immunostaining with CK-18 and CK-19 and their positivity in GOC may further help in differentiating GOC from central mucoepidermoid carcinoma (CMEC). Certain studies demonstrated that the use of immunohistochemistry (IHC) for p-53 and Ki-67 can help in differentiating GOC from CMEC. GOC exhibited decreased p-53 positivity and increased Ki-67 index when compared to CMEC. 
It is of interest to note that in this case, the cyst was bilateral and in the posterior maxillary region, which is seldom encountered; therefore, it is always necessary to distinguish GOC from surgical ciliated cyst. In this case, negative surgical history in the same area ruled out the possibility of surgical ciliated cyst and favored the diagnosis of GOC.
Several treatment modalities have been used. These include curettage, enucleation with careful dissection of the margins and local block excision. The prognosis of this cyst still remains unclear. However, the aggressive nature of the lesion has been reported, and at least 25 to 55% recur following curettage. It is imperative to follow up the patient carefully for recurrent lesion several years after curettage or enucleation, since cases have been reported as long as 7 years after original treatment. 
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Vikram S Amberkar
Department of Oral Pathology & Microbiology, Bapuji Dental College and Hospital, Davanagere, Karnataka
Source of Support: None, Conflict of Interest: None
[Figure 1], [Figure 2], [Figure 3], [Figure 4], [Figure 5], [Figure 6]