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An Official Publication of the Indian Association of Oral and Maxillofacial Pathologists


 
  Table of Contents    
CASE REPORT  
Year : 2014  |  Volume : 18  |  Issue : 2  |  Page : 320-323
 

Glandular odontogenic cyst in maxilla: A case report and literature review


1 Department of Oral Pathology and Microbiology, Jodhpur Dental College and General Hospital, Boranada, Rajasthan, India
2 Department of Oral Pathology and Microbiology, K. M. Shah Dental College and Hospital, Vadodara, Gujarat, India
3 Department of Dentistry, Dr. Sampurnanand Medical College and Hospital, Jodhpur, Rajasthan, India

Date of Submission18-Jul-2013
Date of Acceptance07-Aug-2014
Date of Web Publication17-Sep-2014

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Source of Support: None, Conflict of Interest: None


DOI: 10.4103/0973-029X.140923

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   Abstract 

Glandular odontogenic cyst (GOC) is an uncommon jaw bone cyst of odontogenic origin described in 1987 by Gardner et al. It is a cyst having an unpredictable and potentially aggressive behavior. The increased recurrence rate can be due to its multilocularity and incomplete removal of the lining following conservative treatment. Clinically, the most common site of occurrence is the anterior region of mandible. GOC has a slight male predilection and occurs primarily in middle-aged patients. This article presents a case of glandular odontogenic cyst in a 30-year-old female patient in the posterior region of the maxilla, which is quite rare.


Keywords: Glandular cyst, maxilla, odontogenic cyst


How to cite this article:
Purohit S, Shah V, Bhakhar V, Harsh A. Glandular odontogenic cyst in maxilla: A case report and literature review. J Oral Maxillofac Pathol 2014;18:320-3

How to cite this URL:
Purohit S, Shah V, Bhakhar V, Harsh A. Glandular odontogenic cyst in maxilla: A case report and literature review. J Oral Maxillofac Pathol [serial online] 2014 [cited 2019 Nov 20];18:320-3. Available from: http://www.jomfp.in/text.asp?2014/18/2/320/140923



   Introduction Top


Glandular odontogenic cyst (GOC) is a rare lesion that arises in the tooth bearing areas of the jaws. Padayachee and Van Wyk initially reported it as a sialodontogenic cyst in 1987 based on the possibility of salivary gland origin but its odontogenic origin was first described in 1988 by Gardner et al., who proposed the name GOC because the cyst wall epithelium was odontogenic and contained mucin elements with no evidence of salivary tissue involvement. [1],[2],[3] The term mucoepidermoid cyst or mucous producing cyst was used by Sadeghib in 1991 due to the microscopic findings of mucus producing cells and squamous cells. [4],[5] In the 1992 World Health Organization (WHO) typing of odontogenic tumors, GOC was defined as "a cyst arising in the tooth-bearing areas of the jaws characterized 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". [6]

GOC is relatively rare lesion with a frequency rate of 0.012-1.3% of all the jaw cysts and its prevalence rate is 0.17%. [7] It has two clinically important attributes: A "high recurrence rate'' and an "aggressive growth potential''. [8]

GOC primarily occurs in middle-aged patients with slight male predilection and the most common site of occurrence is mandibular anterior region where it usually presents as a painless, slow-growing swelling. Radiographically, these cysts are described as well-defined, unilocular or multilocular without specific diagnostic characteristics. [9] Histologically, GOC shows a non-keratinized stratified squamous epithelial lining, focal plaque like thickenings within the lining, microcysts or intraepithelial crypts containing mucin, mucous cells and hyaline bodies, eosinophilic cuboidal or columnar cells that may be ciliated, papillary projections of epithelium and absence of inflammation in the subepithelial connective tissue. [1],[5],[10],[11] The relative rarity of the lesion prompted us to add one more of our case and review the literature.


   Case report Top


A 30-year-old female patient reported with a swelling in the upper right back region of the jaw. The swelling was present since 8 months which was painless and increased gradually in size. Extraoral examination showed slight asymmetry with fullness of the right side of the face. Intraorally a diffuse, non fluctuant and firm swelling was seen with normal overlying mucosa extending from the buccal aspect of 11-17 obliterating the vestibule [Figure 1]. The teeth were tender on percussion . Panoramic radiographic examination revealed a well-defined unilocular radiolucency extending from 15-17. 16 was carious with root resorption and roots of 15 and 17 were displaced [Figure 2]. A provisional diagnosis of radicular cyst was made. Gross examination showed a smooth to rough mass measuring 2.5 × 2 cm and the cut section showed a cystic wall filled with necrotic material [Figure 3]. Histopathologic examination, revealed a cystic cavity lined by a non keratinized stratified squamous epithelium with surface layer composed of ciliated columnar cells [Figure 4] and [Figure 5]. Intraepithelial crypts containing PAS positive mucous cells were also observed [Figure 6]. Plaque like epithelial thickenings were also seen in one area. Subepithelial inflammatory infiltrate was found in the underlying connective tissue. Immunohistochemistry (IHC) staining was done using CK-18, CK-19, p53, Ki-67. Among them CK-19 and Ki-67 were found positive. [Figure 7] and [Figure 8]. A final diagnosis of GOC was made.
Figure 1: Clinical photograph shows swelling in the upper right back region of the jaw

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Figure 2: Orthopantomogram shows radiolucent area in 15, 16, 17 region

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Figure 3: Gross specimen showing a smooth lobulated mass

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Figure 4: Photomicrograph shows lining epithelium (H&E stain, ×100)

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Figure 5: Photomicrograph shows surface ciliated columnar epithelium (H&E stain, ×400)

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Figure 6: Photomicrograph shows special stained mucous cells (PAS stain, ×400)

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Figure 7: Photomicrograph shows strong positive immunoreactions to the Ki-67 protein (IHC stain, ×100)

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Figure 8: Photomicrograph shows strong positive immunoreactions to the CK-19 protein (IHC stain, ×100)

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   Discussion Top


GOC is a relatively rare entity. Magnusson and co-authors evaluated 5900 cases of jaw bone cysts and found only 7 cases of GOC, about 0.12%; whereas Van Heerden and others reported 1.3% of GOC in their study. [7],[12]

In the present case the patient is a middle-aged female whereas existing literature reports a slight male predilection. The most common site of occurrence is the anterior region of mandible followed by anterior region of maxilla and posterior region of mandible. [2],[11],[13],[14] Occurrence of GOC in posterior region of maxilla is rare. About three cases have been reported till date and ours is probably the fourth case.

A diagnosis based only on clinical and radiological examination is difficult because of similarities with various other intrabony pathologies, hence a histopathological evaluation becomes mandatory. [15]

The histopathological characteristics of GOC have been divided into major and minor categories by Kaplan et al. [16] GOC should be distinguished from lateral periodontal cyst, botryoid odontogenic cyst, surgical ciliated cyst, radicular cyst with mucous metaplasia and central MEC as it exhibits considerable overlapping of histopathological features [Figure 9]. [17],[18]
Figure 9: Histopathological differential diagnosis of glandular odontogenic cyst17

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Kaplan et al., in their study found that GOC showed positivity for p53 and Ki-67. When compared to MEC, these markers could be auxillary aids in the differential diagnosis of these lesions. According to various authors, positive immunostaining with CK-18 and CK-19 in GOC may further help in differentiating GOC from central mucoepidermoid carcinoma (MEC). A recent study found significantly higher expression of both cytoplasmic and nuclear MASPIN in the mucous cells in low grade MEC (16.5% cytoplasmic, 1.7% nuclear) as compared with GOC (1.5% and 0.3%) or odontogenic cysts with mucous metaplasia (1% and 0.4%). [17],[18]

Several treatment modalities have been used which 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 the recurrence rate is directly related to the size of the lesion. 14.4% of the small lesions recur in contrast to 85.6% of the large lesions. [16] Therefore, large lesions should be treated more aggressively and followed for a long period. [17],[19]


   Conclusion Top


GOC is a rare cyst in maxillary region. It is important to consider histopathological features for its diagnosis since it bears resemblance to lesions like MEC. IHC provides an additional tool for its differential diagnosis.

 
   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.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. 2
    
3.Ambekar VS, Jahagirdar A, Ahmed Mujib BR. Glandular odontogenic cyst: Report of an unusual bilateral occurance. Indian J Dent Res 2011;22:364.  Back to cited text no. 3
    
4.Lin CC, Chen CH, Lai S, Chen YK, Wan WC, Lu SY, et al. Glandular odontogenic cyst: A case report. Kaohsiung J Med Sci 2000;16:53-8.  Back to cited text no. 4
    
5.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. 5
    
6.Macdonald-Jankowski DS. Glandular odontogenic cyst: Systematic review. Dentomaxillofac Radiol 2010;39:127-39.  Back to cited text no. 6
[PUBMED]    
7.Magnussson B, Goransson L, Odesjo B, Grondahl K, Hirsch JM. Glandular odontogenic cyst. Report of seven cases. Dentomaxillofac Radiol 1997;26:26-31.  Back to cited text no. 7
    
8.Shen J, Fan M, Chen X, Wang S, Wang L, Li Y. Glandular odontogenic cyst in China: Report of 12 cases and immunohistochemical study. J Oral Pathol Med 2006;35:175-82.  Back to cited text no. 8
    
9.Nofke C, Raubenheimer EJ. The glandular odontogenic cyst: Clinical and radiological features; review of the literature and report of nine cases. Dentomaxillofac Radiol 2002;31:333-8.  Back to cited text no. 9
    
10.Nair G, Varghese V, Shameena M, Sudha S. Glandular odontogenic cyst: Report of a case and review of literature. J Oral Maxillofac Pathol 2006;10:20-3.  Back to cited text no. 10
  Medknow Journal  
11.Salehinejad J, Sagha S, Zare-Mahmoodabadi R, Ghazi N, Kermani H. Glandular odontogenic cyst of the posterior maxilla. Arch Iran Med 2011;14:416-8.  Back to cited text no. 11
    
12.van Heerden WF, Raubenheimer EJ, Turner ML. Glandular odontogenic cyst. Head Neck 1992;14:316-20.  Back to cited text no. 12
    
13.Kasaboglu O, Basal Z, Usubutun A. Glandular odontogenic cyst presenting as a dentigerous cyst: A case report. J Oral Maxillofac Surg 2006;64:731-3.  Back to cited text no. 13
    
14.Sittitavornwong S, Koebler JR, Said-Al-Naief N. Glandular odontogenic cyst of the anterior maxilla: Case report and review of literature. J Oral Maxillofac Surg 2006;64:740-5.  Back to cited text no. 14
    
15.Guruprasad Y, Chauhan DS. Glandular odontogenic cyst of maxilla. J Clin Imaging Sci 2011;1:54.  Back to cited text no. 15
    
16.Kaplan I, Gal G, Anavi Y, Manor R, Calderon S. Glandular odontogenic cyst: Treatment and recurrence. J Oral Maxillofac Surg 2005;63:435-41.  Back to cited text no. 16
    
17.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. 17
    
18.Prabhu S, Rekha K, Kumar G. Glandular odontogenic cyst mimicking central mucoepidermoid carcinoma. J Oral Maxillofac Pathol 2010;14:12-5.  Back to cited text no. 18
[PUBMED]  Medknow Journal  
19.Stoelinga PJ. The management of aggressive cysts of the jaws. J Maxillofac Oral Surg 2012;11:2-12.  Back to cited text no. 19
[PUBMED]    


    Figures

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



 

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