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 Table of Contents  
CASE REPORT
Year : 2017  |  Volume : 3  |  Issue : 1  |  Page : 40-45

Glandular odontogenic cyst: Analysis of clinicopathological features of five cases


1 Department of Oral and Maxillofacial Pathology, D.A. Pandu Memorial R. V. Dental College and Hospital, Bengaluru, Karnataka, India
2 Department of Oral and Maxillofacial Surgery, D.A. Pandu Memorial R. V. Dental College and Hospital, Bengaluru, Karnataka, India

Date of Web Publication17-Jul-2017

Correspondence Address:
K K Deepa
Department of Oral and Maxillofacial Pathology, D.A. Pandu Memorial R. V. Dental College and Hospital, Bengaluru, Karnataka
India
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Source of Support: None, Conflict of Interest: None


DOI: 10.4103/ijohr.ijohr_22_17

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  Abstract 

Glandular odontogenic cyst (GOC) is a rare developmental cystic lesion of the jaws accounting for 0.012%–0.3% of all the odontogenic cysts. It occurs most commonly in middle-aged men, especially in the anterior mandible. It clinically presents as a slow-growing intraosseous lesion. Small cysts may be asymptomatic while larger ones may cause expansion with pain or paresthesia. Radiographically, it frequently presents as a multilocular radiolucency. Microscopic features are characterized by nonkeratinized stratified squamous epithelium of variable thickness with many microcystic or pseudoglandular spaces and plaque-like extensions. Although its microscopic features are characteristic, few situations may arise where this entity may be confused with dentigerous, botryoid odontogenic, radicular cyst, and central mucoepidermoid carcinoma. The importance of GOC relates to its high rate of recurrence making its diagnosis important. The purpose of this study is to present the clinicopathological features of five cases of GOC which have been reported in our institution.

Keywords: Glandular odontogenic cyst, microcysts, odontogenic cyst


How to cite this article:
Yanduri S, Deepa K K, Kumar B V, Suma S, Madhura M G, Vakade CD. Glandular odontogenic cyst: Analysis of clinicopathological features of five cases. Indian J Oral Health Res 2017;3:40-5

How to cite this URL:
Yanduri S, Deepa K K, Kumar B V, Suma S, Madhura M G, Vakade CD. Glandular odontogenic cyst: Analysis of clinicopathological features of five cases. Indian J Oral Health Res [serial online] 2017 [cited 2022 May 18];3:40-5. Available from: https://www.ijohr.org/text.asp?2017/3/1/40/210923


  Introduction Top


Glandular odontogenic cyst (GOC) is a rare odontogenic cyst of the jaw bones, accounting for 0.012%–0.3% of all odontogenic cysts.[1] It usually presents as a slow-growing, asymptomatic swelling generally affecting the anterior parts of jaws, particularly the mandible. Radiographically, GOC is localized intraosseously and may appear as a multilocular or unilocular radiolucent lesion with well-defined borders. Although the histopathological features are unique, situations may arise wherein similar features may be seen in other cysts or even tumors, thus posing a challenge in making the diagnosis.[2],[3],[4] GOC has an aggressive potential with a relatively high rate of recurrence.[5] Therefore, the correct diagnosis is a major challenge and is of extreme clinical importance. This study for the first time emphasizes on the correlation of histopathological criteria given by two different authors, which has to be followed while diagnosing the cases of GOC.

The aim of the present study is to discuss the histopathological features and the differential diagnosis of five cases of GOC reported in our institution.


  Report of Five Cases Top


Five cases of GOC were diagnosed over 7 years (2009–2015) in the Department of Oral and Maxillofacial Pathology, DA Pandu Memorial RV Dental College, Bengaluru, Karnataka. Details of clinical data, radiographic features, and provisional diagnosis are shown in [Table 1].
Table 1: Clinical data, radiographic features, and provisional diagnosis of five cases

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Clinical and radiographic data

The age of the patients ranged from 13 to 60 years with a mean of 41.5 years. Three females and 2 males were affected. The most common site was the mandibular posterior region (three cases) with the remaining two cases occurring in the maxillary anterior region. Only one case was symptomatic. Radiographically, all patients presented with a unilocular radiolucency [Figure 1] and [Figure 2].
Figure 1: Case 3: Unilocular radiolucency (arrow) in the anterior region of the maxilla with expansion and destruction of cortical plates (cone-beam computed tomography)

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Figure 2: Case 5: Unilocular radiolucency (arrow) seen on the posterior part of the right mandible associated with an impacted tooth

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Histopathological data

Histopathological data were analyzed by using two methodologies: One was with the criteria given by Kaplan et al. [Table 2], and the second by Fowler et al. [Table 3].[6],[7]
Table 2: Correlation of histopathological features of the five reported cases with the major and minor criteria given by Kaplan et al.

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Table 3: Correlation of histopathological features of the 5 reported cases with the parameters given by Fowler et al.

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Kaplan et al. divided the histopathological features diagnostic of GOC into major and minor criteria. Compatibility with all major criteria was required for a diagnosis while the minor criteria played a supportive role.[6] All the five cases in the case series fulfilled the major criteria.

Fowler et al. combined all the histopathological features into ten parameters and suggested that seven or more parameters need to be met for a diagnosis of GOC.[7] In the present case series, four cases met more than seven criteria while one case fulfilled six criteria.

The important histopathological features of all the 5 cases are illustrated in [Figure 3], [Figure 4], [Figure 5], [Figure 6], [Figure 7], [Figure 8], [Figure 9], [Figure 10]. Other features such as inflammation (two cases), cholesterol clefts (two cases), calcifications (two cases), and epithelial rests (one case) were also seen.
Figure 3: Squamous epithelium lining of variable thickness (blue arrow) with a flat interface with the connective tissue wall (orange arrow) (H and E, ×40)

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Figure 4: Cuboidal eosinophilic cells or hobnail cells (arrow) (H and E, ×200)

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Figure 5: Mucous (arrow) cells seen within the cyst lining (periodic acid-Schiff, ×40)

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Figure 6: Intraepithelial pseudoglandular, microcystic spaces (arrow) lined by cuboidal cells (H and E, ×40)

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Figure 7: Papillary projection of the epithelium (arrow) (H and E, ×40)

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Figure 8: Ciliated cells (arrow) seen in the epithelial lining (H and E, ×40)

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Figure 9: Multicystic or multiluminal compartments (arrow) (H and E, ×40)

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Figure 10: Clear cells (arrow) in the spinous layer of the epithelium (H and E, ×40)

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  Discussion Top


GOC was first documented as a sialo-odontogenic cyst by Padayachee and Van Wyk in 1988.[8] Gardner et al. in 1988 characterized the histopathological features of this entity and proposed the term GOC.[9] In 1992, the WHO accepted the term GOC and categorized it under the group of developmental odontogenic cysts.[10]

GOC has been reported to occur in patients older than 30 years of age with a male predilection. In the present case series, three patients were female and two were male. The prominent location of GOC in this study was in the mandibular region which correlates with that of literature.

GOC is a cyst of multiple histopathological features; however, often all the features are seen only at focal areas or not all the features are seen in the cyst. In addition, few of these features are known to occur in other lesions such as botryoid odontogenic cyst, radicular cyst (RC), dentigerous cyst, and low-grade mucoepidermoid carcinoma. Therefore, this may make arriving at a diagnosis difficult.[11]

To address this problem, Kaplan et al. put forward a set of criteria for histological diagnosis wherein all the histological features were divided into major and minor criteria. They suggested that focal presence of all major criteria needs to be met, while the minor ones support the diagnosis.[6] In the present study, all the five cases met all the major criteria and a few of the minor criteria, therefore confirming the diagnosis of GOC.

However, Fowler et al. believed that the presence of all of the major criteria does not need to be met for a diagnosis. They proposed a combination of ten specific microscopic features and said that the presence of seven or more microscopic findings was highly predictive of a diagnosis of GOC. They suggested that when five or less parameters were met, the diagnosis may be that of non-GOC or what they called as a “GOC mimicker.”[7] In the present study, four cases met with more than seven criteria, with only one case meeting six criteria. As a result of this, all can be considered as GOCs.

In the present case series, two cases were given a provisional diagnosis of dentigerous cyst because of the presence of a pericoronal radiolucency. Often, metaplastic changes in the epithelial lining of dentigerous cysts such as the presence of ciliated cells, mucous cells, and eosinophilic cuboidal cells may mimic a GOC.[12],[13],[14] Similar histopathological changes may also be seen in RCs, especially ciliated cells which are evident in cases of RCs of the maxilla.[12],[13],[14] In one of the present cases, the cyst was associated with a nonvital tooth as well as showed areas of intense inflammatory infiltrate with the epithelium exhibiting an arcading pattern simulating an RC. In addition, cholesterol clefts were also seen. In addition, one case had multicystic compartments which may be confused with that of a botryoid odontogenic cyst.

Another important differential diagnosis is that of a low-grade mucoepidermoid carcinoma (MEPCa) due to the presence of multiple cyst-like structures lined by mucus-producing cells, clear cells, and epidermoid component which are typically found.[15],[16]

However, the fact that all these cysts in the present case series had the additional features of a variable thickness of the epithelial lining, epithelial spheres, microcysts with periodic acid–Schiff-positive material, crypt formation, and clear cells helped us arrive at a diagnosis of GOC. According to Fowler et al., the above-mentioned set of factors are supposed to be most helpful in distinguishing GOCs from GOC mimickers.[7] The problem may be further magnified in cases where an incisional biopsy is received and the characteristic features are seen only at focal areas. In such situations, it is recommended that the report contains a mention of the fact that GOC-like features are seen so that the surgeon can accordingly plan the treatment.

Kaplan et al. investigated the use of p53, Ki67, and PCNA as an aid in the diagnosis of GOC.[6] Mean p53 labeling index (LI) was higher in GOC and MEPCa than in RC with mucous metaplasia (RCM) which showed a very minimal expression. They suggested that the expression of p53 in GOC though higher than RC is not as high as that of squamous cell carcinoma. Ki67 LI was higher in GOC and RCM than in low-grade MEPCa, thus indicating the indolent behavior of this salivary gland tumor. These molecular markers can help in diagnosing GOC in situ ations of overlapping histopathology.[6]

There was a time in literature when this cyst was speculated to be of salivary gland origin due to the presence of numerous mucous-producing cells. In fact, Sadeghi in 1991 termed this cyst as a mucoepidermoid odontogenic cyst or mucous-producing cyst.[17] Although studies on the cytokeratin profile failed to support either an odontogenic origin or a sialogenic origin, there is ample evidence to favor the former.[18] Histologically, the presence of odontogenic epithelial plaques and whorls which are also seen in other odontogenic cysts and the fact that this cyst has been reported to occur along with other odontogenic tumors such as ameloblastoma and keratocystic odontogenic tumor have confirmed its odontogenic nature. The minimal expression of mammary serine protease inhibitor and epithelial membrane antigen in GOC as opposed to the significantly higher expression in MEPCa is an additional indication against a true glandular origin, in spite of the morphologic resemblance.[19] In our study, we also found that one particular case showed evidence of epithelial rests in the connective tissue capsule which further confirms its odontogenic origin.

Treating GOC ranges from enucleation for small unilocular lesions to marginal resection and en bloc excision for large lesions. The choice of treatment thus depends on the size of the lesion, integrity of the jaw borders, and proximity of the lesion to vital structures.[5] A global recurrence rate of 18% has been reported with the risk of recurrence depending on the size, presence of multilocularity, and choice of treatment.[20] The follow-up period for patients has been recommended to be a minimum of 3 years and preferably up to 7 years.


  Conclusion Top


GOC is a rare lesion with unique microscopic features. If all the histopathological features are evident, then the diagnosis is straightforward. The problem arises with GOC mimickers in which situations it is recommended to use criteria suggested by either Kaplan et al. or Fowler et al. to arrive at a diagnosis.

Financial support and sponsorship

Nil.

Conflicts of interest

There are no conflicts of interest.

 
  References Top

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Manzini M, Deon C, Corte LD, José Bertotto C, de Abreu LB. Glandular odontogenic cyst: An uncommon entity. Rev Bras Otorrinolaringol 2009;75:320.  Back to cited text no. 1
    
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Mittal A, Narang V, Kaur G, Sood N. Glandular Odontogenic Cyst of Mandible: A Rare Entity 2015;9:ED09-10.  Back to cited text no. 3
    
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Fowler CB, Brannon RB, Kessler HP, Castle JT, Kahn MA. Glandular odontogenic cyst: Analysis of 46 cases with special emphasis on microscopic criteria for diagnosis. Head Neck Pathol 2011;5:364-75.  Back to cited text no. 7
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Padayachee A, Van Wyk CW. Two cystic lesions with features of both the botryoid odontogenic cyst and the central mucoepidermoid tumour: Sialo-odontogenic cyst? J Oral Pathol 1987;16:499-504.  Back to cited text no. 8
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Kramer IR, Pindborg JJ, Shear M. The WHO histological typing of odontogenic tumours: A commentary on the second edition. Cancer 1992;70:2988-94.  Back to cited text no. 10
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Shah AA, Sangle A, Bussari S, Koshy AV. Glandular odontogenic cyst: A diagnostic dilemma. Indian J Dent 2016;7:38-43.  Back to cited text no. 11
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Takeda Y, Oikawa Y, Furuya I, Satoh M, Yamamoto H. Mucous and ciliated cell metaplasia in epithelial linings of odontogenic inflammatory and developmental cysts. J Oral Sci 2005;47:77-81.  Back to cited text no. 13
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Kaplan I, Shuster A, Reiser V, Shlomi B. Histologic look-a likes: Glandular odontogenic cyst and odontogenic cysts with mucous metaplasia. JSM Dent Surg 2016;1:1005.  Back to cited text no. 14
    
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Pires FR, Chen SY, da Cruz Perez DE, de Almeida OP, Kowalski LP. Cytokeratin expression in central mucoepidermoid carcinoma and glandular odontogenic cyst. Oral Oncol 2004;40:545-51.  Back to cited text no. 16
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    Figures

  [Figure 1], [Figure 2], [Figure 3], [Figure 4], [Figure 5], [Figure 6], [Figure 7], [Figure 8], [Figure 9], [Figure 10]
 
 
    Tables

  [Table 1], [Table 2], [Table 3]


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