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 Table of Contents  
CASE REPORT
Year : 2021  |  Volume : 26  |  Issue : 2  |  Page : 129-131

Glandular odontogenic cyst: A case report of an unusual lesion


1 Department of Oral Pathology, SRM Dental College, Chennai, Tamil Nadu, India
2 Chief Consultant, Rema's Dental Clinic, Trivandrum, Kerala, India

Date of Submission10-Nov-2020
Date of Acceptance30-Jun-2021
Date of Web Publication10-Feb-2022

Correspondence Address:
Dr. Nandhini Gunasekaran
Department of Oral Pathology, SRM Dental College, Ramapuram, Chennai, Tamil Nadu
India
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Source of Support: None, Conflict of Interest: None


DOI: 10.4103/jmgims.jmgims_133_20

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  Abstract 


Glandular odontogenic cyst (GOC) is a rare cyst occurring in the jaw bone having an uncertain and aggressive behavior with high recurrence rate. The diagnosis of this lesion is trying as it can be confused with other jaw cysts and malignant lesions. Treatment methods vary from conservative surgical treatment to jaw resection. This article presents a case of GOC mimicking radiographically as a lateral periodontal cyst.

Keywords: Hobnail cells, intraepithelial crypt, maxilla, mucous cells, pseudoglandular


How to cite this article:
Gunasekaran N, Krishnan R, Krishnan R. Glandular odontogenic cyst: A case report of an unusual lesion. J Mahatma Gandhi Inst Med Sci 2021;26:129-31

How to cite this URL:
Gunasekaran N, Krishnan R, Krishnan R. Glandular odontogenic cyst: A case report of an unusual lesion. J Mahatma Gandhi Inst Med Sci [serial online] 2021 [cited 2022 May 24];26:129-31. Available from: https://www.jmgims.co.in/text.asp?2021/26/2/129/337460




  Introduction Top


Glandular odontogenic cyst (GOC) is a rare cyst of the jaws. It was first termed as “sialo-odontogenic cyst” in 1987 by Padayachee and van Wyk.[1] Later in 1988, Gardner et al. in 1988 endorsed the term GOC,[2] which was further classified as a developmental type of odontogenic cyst by the WHO in 1992.[3]

GOC is uncommon, accounting for 0.012%–1.3% of all jaw bone cysts and to date, only 220 cases have been globally reported from 1987.[4] It is commonly seen in the anterior region of the mandible, and appears as an asymptomatic swelling but with a potentially aggressive behavior and increased recurrence rate.

This report documents an uncommon case of GOC in the maxilla of a 17-year-old boy which resembled a lateral periodontal cyst in the radiograph and was treated with enucleation.


  Case Report Top


A 17-year-old boy reported to our institution with complaints of discomfort and swelling in the anterior region of the upper jaw for the past 7 months duration. The patient reported that the swelling was slow-growing and painless. He was not able to remember the time of onset of the swelling. The patient's medical history was not significant.

Extraorally no facial asymmetry was seen. Intraoral examination revealed a solitary swelling measuring 1 cm × 1.5 cm with diffuse margins in the facial surface of the right maxilla between number 13 and 14. The mucosa overlying the swelling was of normal color and on palpation the swelling was firm, nontender, nonpulsatile, and immobile [Figure 1]. The teeth adjacent to the swelling were subjected to electronic pulp testing and were found to be vital.
Figure 1: Intraoral picture depicting solitary swelling between 13 and 24 with diffused margins

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Radiograph examination revealed a well-defined unilocular radiolucency with sclerotic border between the roots of teeth number 13 and 14 with root divergence [Figure 2]. There was no evidence of root resorption.
Figure 2: Intraoral periapical exhibiting unilocular radiolucency with well-defined borders

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Based on the clinical and radiographic appearances, a provisional diagnosis of the lateral periodontal cyst was made.

The patient was subjected to routine blood investigations and it was found to be within the normal limits. Under local anesthesia, the lesion was surgically enucleated and sent for histopathological examination.

The microscopic examination of the hematoxylin and eosin-stained section revealed a cystic cavity lined by nonkeratinized stratified squamous epithelium and underlying connective tissue wall [Figure 3]. The lining epithelium exhibited pseudoglandular structures, intraepithelial crypts, and microcyst formation [Figure 4].
Figure 3: Nonkeratinized lining epithelium with pseudoglandular and microcystic areas (Hematoxylin and eosin, 100×)

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Figure 4: Pseudoglandular areas (Hematoxylin and eosin, 200×)

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Based on the history, clinical, radiographic, and histopathological findings, the final diagnosis of GOC was made.

So far, on 3-year follow-up, no recurrence was observed and intraoral periapical radiographs revealed new bone formation at the site of the cystic lesion on the follow-up visits [Figure 5].
Figure 5: Follow-up intraoral periapical showing new bone formation

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  Discussion Top


GOC is an uncommon odontogenic cyst which is locally aggressive with high recurrence potential. The origin of the lesion is still uncertain, with some authors suggesting that it develops from the intraosseous salivary gland tissue. More recently it is thought to originate from the odontogenic epithelium.[5]

It usually occurs within a wide age range of 14–75 years and very rarely seen in children. It has a slight male predilection with the male: female ratio being 3:1. The majority of the lesions usually occur in the anterior region of the mandible (72%). In the case of maxilla, it is usually seen in the canine region (28%), which was same in our case.[6] The cyst commonly presents with swelling with (28.5%) or without pain (71.5%). Pain in GOC may be due to secondary infection or compression of the associated neurovascular bundle. In some cases, it is usually discovered as an incidental radiographic finding.[7]

Radiographically, the lesion presents as either unilocular (76.9%) or multilocular (23.1%) radiolucency with well-defined or scalloped osteosclerotic borders and sometimes associated with root resorption and root displacement.[7] Radiographically, it mimics a wide range of lesions such as lateral periodontal cysts, botryoid cysts, residual cysts, odontogenic keratocysts, ameloblastoma, and central mucoepidermoid carcinoma.[6] In our case, the lesion radiographically mimicked lateral periodontal cyst.

Histopathologically for the diagnosis of GOC, Kaplan et al. have categorized the microscopic features into major and minor criteria.[8] However, for confirmatory diagnosis, major criteria must be present, whereas minor criteria are not mandatory but could be supportive for the diagnosis.

The major criteria include:

  1. Nonkeratinized squamous epithelial lining with a flat interface with the connective tissue wall
  2. Varying thickness of epithelial lining with or without epithelial spheres or epithelial whorls or focal proliferation
  3. Cuboidal eosinophilic cells or hobnail cells in lining epithelium
  4. Mucous cells showing intraepithelial mucous pools, with or without crypts
  5. Intraepithelial glandular microcystic or duct-like structures.


The minor criteria include:

  1. Lining epithelium exhibiting papillary proliferation
  2. Ciliated cells
  3. Multicystic or multiluminal architecture
  4. Clear or vacuolated cells in basal or spinous cell layers.


Thus, the present case showed some of Kaplan's histopathological criteria, such as nonkeratinized stratified squamous epithelium, pseudoglandular structures, intraepithelial crypt, and microcysts formation for confirming the diagnosis of GOC.

It is important to distinguish GOC histopathologically from low-grade mucoepidermoid carcinoma. It can be differentiated by the presence of intraepithelial microcyst or duct-like structures, superficial cuboidal cells, ciliated cells, epithelial whorls, which is not seen in low-grade mucoepidermoid carcinoma.[7]

The treatment of GOC is controversial ranging from curettage, enucleation, en bloc, and partial osteotomy due to its high recurrence rate.[7] Follow-up for at least 3–7 years is recommended for this lesion.


  Conclusion Top


GOC is a rare lesion and difficult to diagnose due to its similarities with a variety of lesions. Hence, proper clinical and radiographic evaluation should be done along with careful microscopic evaluation. Complete surgical removal with long-term follow-up should be advocated to prevent a recurrence.

Declaration of patient consent

The authors certify that they have obtained all appropriate patient consent forms. In the form the patient(s) has/have given his/her/their consent for his/her/their images and other clinical information to be reported in the journal. The patients understand that their names and initials will not be published and due efforts will be made to conceal their identity, but anonymity cannot be guaranteed.

Financial support and sponsorship

Nil.

Conflicts of interest

There are no conflicts of interest.



 
  References Top

1.
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. 1
    
2.
Gardner DG, Kessler HP, Morency R, Schaffner DL. The glandular odontogenic cyst: An apparent entity. J Oral Pathol 1988;17:359-66.  Back to cited text no. 2
    
3.
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. 3
    
4.
Krishnamurthy A, Sherlin HJ, Ramalingam K, Natesan A, Premkumar P, Ramani P, et al. Glandular odontogenic cyst: Report of two cases and review of literature. Head Neck Pathol 2009;3:153-8.  Back to cited text no. 4
    
5.
Mascitti M, Santarelli A, Sabatucci A, Procaccini M, Lo Muzio L, Zizzi A, et al. Glandular odontogenic cyst: Review of literature and report of a new case with cytokeratin-19 expression. Open Dent J 2014;8:1-12.  Back to cited text no. 5
    
6.
Faisal M, Ahmad SA, Ansari U. Glandular odontogenic cyst-Literature review and report of a paediatric case. J Oral Biol Craniofac Res 2015;5:219-25.  Back to cited text no. 6
    
7.
Ferreira JC, Vêncio EF, de Sá RT, Gasperini G. Glandular odontogenic cyst in dentigerous relationship: An uncommon case report. Case Rep Dent 2019;2019:8647158.  Back to cited text no. 7
    
8.
Kaplan I, Anavi Y, Hirshberg A. Glandular odontogenic cyst: A challenge in diagnosis and treatment. Oral Dis 2008;14:575-81.  Back to cited text no. 8
    


    Figures

  [Figure 1], [Figure 2], [Figure 3], [Figure 4], [Figure 5]



 

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