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


 
CASE REPORT Table of Contents   
Year : 2006  |  Volume : 10  |  Issue : 1  |  Page : 20-23
 

Glandular odontogenic cyst: Report of a case and review of literature


Department of Oral Pathology, Govt. Dental College, Calicut 673008, India

Correspondence Address:
R G Nair
Department of Oral Pathology, Govt. Dental College, Calicut 673008
India
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Source of Support: None, Conflict of Interest: None


DOI: 10.4103/0973-029X.37791

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   Abstract 

The glandular odontogenic cyst (GOC) is a recently recognized rare developmental odontogenic cyst showing an aggressive behavior and accounts for 0.012 to 1.3% of all jaw cysts. This cyst is possibly derived from rests of dental lamina and comprises both secretory elements and stratified squamous epithelium. Many authors have suggested that the cyst mainly occurs in 4th and 5' decades in the mandible and presents as an expansion with or without pain or Paraesthesia. The treatment of choice is excision rather than enucleation in order to obviate recurrence. Here, we report a case of GOC in a 45-year-old female patient, in the posterior region of the maxilla, which is quite rare.


Keywords: Glandular Odontogenic cyst; Sialo-Odontogenic Cyst.


How to cite this article:
Nair R G, Varghese I V, Shameena P M, Sudha S. Glandular odontogenic cyst: Report of a case and review of literature. J Oral Maxillofac Pathol 2006;10:20-3

How to cite this URL:
Nair R G, Varghese I V, Shameena P M, Sudha S. Glandular odontogenic cyst: Report of a case and review of literature. J Oral Maxillofac Pathol [serial online] 2006 [cited 2019 Nov 18];10:20-3. Available from: http://www.jomfp.in/text.asp?2006/10/1/20/37791



   Introduction Top


Glandular odontogenic cyst was first described by Padayachee and Van Wyk in 1987[1]. It is a developmental jaw cyst with characteristic histopathological features and biological behavior and established as a distinct entity by Gardner et al in 1988 [2]. The lesion is also described as (a) sialo-odontogenic cyst [1] (b) Mucoepidermoid odontogenic cyst [3] and (c) polymorphous odontogenic cyst [4],[5].

The histogenesis of this lesion is unclear. Initially it was suggested to originate from intra osseous salivary gland tissue but recently an odontogenic origin is suggested because of histopathological features and cytokeratin profile[5].

Glandular odontogenic cysts are rare and occur in a wide age range; the mean age being 49.5 years [6]. Middle aged men are commonly affected [7]. They form within the bone and produce painless slow growing swellings. Radiographic examination reveals a well defined unilocular or multilocular radiolucency often with scalloped margins [8]. They can recur if excised inadequately. Eighty eight percent of cysts occur in the anterior mandible and most of them crossed the midline [6].

We herewith present an unusual case of GOC in a 45year-old female patient occurring in the posterior maxilla.


   Case report Top


A 45-year-old female patient reported with the chief complaint of a painful swelling in the right maxilla of 6 months duration. History revealed that the swelling started 6 months back as a small one and gradually increased in size.

Extra orally, the swelling was not obvious and did not produce any facial asymmetry. Intraoral examination revealed a swelling of 2 cm x 2 cm size in 16, 17 and 18 region. The margins were well defined with palatal expansion and obliteration of the buccal vestibule [Figure - 1]. On palpation it was hard and tender. A provisional diagnosis of residual cyst was made. Retention cyst of maxillary antrum and surgical ciliated cyst were considered as the differential diagnoses.

Radiographic examination revealed a radiolucent area in 16, 17, 18 region. [Figure - 2]

CT scan showed small well defined soft tissue density in right maxillary sinus suggestive of cyst/polyp [Figure - 3].

Aspiration yielded clear watery fluid and was submitted for cytological examination which showed sheets of round to cuboidal cells with basophilic nucleus and scanty cytoplasm [Figure - 4]. In some regions spindling can be seen in a dirty background which was suggestive of a salivary gland neoplasm [Figure - 5]. Cyst was enucleated and specimen was submitted for histopathologic examination.

On histological examination the tissue appeared to be part of a cyst, lined by epithelium which is thicker and consists of cuboidal to columnar superficial cells, mucous cells, and ciliated cells. The luminal surface is irregular or papillary. Within the epithelium mucous cells of variable numbers were seen. The connective tissue wall is moderately collagenous with few endothelium lined vascular channels filled with RBCs and focal collections of inflammatory cells [Figure - 6],[Figure - 7]

Special staining revealed the mucous cells to be mucicarmine positive [Figure - 8],[Figure - 9]. The diagnosis of glandular odontogenic cyst was given based on the clinical, radiographic, and histopathologic features.


   Discussion Top


Radiolucent lesions involving the jaws may represent either a developmental disturbance or reactive or inflammatory processes or benign or malignant neoplasms. Irrespective of the cause, these radiolucent lesions should be thoroughly investigated and actively pursued to treat them appropriately.

According to a recent review only less than 50 cases of GOC were reported in English literature [7]. This cyst resembles lateral periodontal cyst, botryoid odontogenic cyst, and in some aspects to Mucoepidermoid carcinoma[8].

Gardner et al, 1988 favoured the term GOC and reported that it can grow to a large size and can recur [9]. Clinically, GOC is generally seen in middle aged persons with the anterior mandibular region being commonly affected. The usual complaints are swelling, pain, or discomfort of the involved area. Radiographically, it appears as a unilocular or multilocular radiolucency with either smooth or scalloped margins.

The cysts are usually lined by [2],[5],[10],[11],[12],[13]

  • An epithelial lining (nonkeratinized stratified squamous epithelium) much thicker than botryoid odontogenic cyst; but also of highly variable thickness.
  • Presents a flat interface with the underlying connective tissue.
  • In more or less extensive areas; the superficial layer of epithelium consists of eosinophilic cuboidal cells, columnar cells, or ciliated cells that form papillary projections and fronds.
  • Pools of mucicarminophilic material, mucous cells, larger granular cells, and vacuolated cells in variable numbers are seen within the epithelium.
  • Within the thickness of epithelium, there are intraepithelial gland like structures consisting of mucous cells and mucin filled crypts or microcysts lined by cuboidal cells that are presumed to result from folding of lining epithelium.
  • Focal thickenings or plaques of epithelium where cells form whorls or spheres are also seen.
  • The sub epithelial connective tissue is usually free of inflammation and may present irregular shaped calcifications or islands of odontogenic epithelium.


Rare findings include an association with ameloblastoma [2] and squamous odontogenic tumor like hyperplasia [14], solid epithelial down growths into the cyst wall [10], satellite microcysts [15], hyaline bodies [16] and epithelial ghost cell calcification [17].

The possible relationship of GOC with intraosseous Mucoepidermoid carcinoma was studied by many authors. GOC shows areas of squamous epithelium containing prominent mucin positive cells and thus resemble in part a Mucoepidermoid carcinoma. But latter is multilocular with cysts lined by flattened squamoid epithelium. This lining also shows foci of proliferation to form whorled nodules or plaques and there may be foci of columnar and ciliated cells. There is also no invasion of surrounding tissues in GOC [13].

GOC has a propensity for recurrence; the frequency of which is estimated to be 21% [5],[7],[8]; appear after the third year. So careful follow up for at least 3 years is recommended [5],[10].

The aggressive biologic behavior and propensity for recurrence might be associated with cell kinetics in the lining epithelium i.e infoldings, microcysts and plaques, which are suggestive of active cell proliferation[5],[12],[18].

Further, immunohistochemical findings in GOC suggest that its biologic behavior may be associated with dysregulation of cell death in the lining epithelium indicated by increased expression of antiapoptotic protein bcl-2. But in spite of its aggressive manner, the cyst frequently recurs; the number of Ki-67 positive cells are lower, which suggest that biologic behavior of GOC is not associated with cell proliferation. Incomplete removal due to its multicystic configuration, tendency of epithelium to separate from connective tissue or growth through cancellous spaces of bone may account for its high recurrence rate [5]

Various treatment modalities are recommended for GOC and suggestions of aggressive enbloc resection and marginal resection were replaced by prevailing view that more conservative methods like excision, enucleation, curettage through extirpation, and cryotherapy should be applied[4],[14],[19].

It is of interest to note that in this case the cyst was in the posterior maxillary region which is often rare. GOC has to be distinguished from surgical ciliated cyst (SCC) and mucosal cyst of antrum. SCC is lined by pseudo stratified ciliated columnar epithelium of respiratory type but can be totally or focally replaced by squamous, cuboidal or columnar cells. Also there may be sub-epithelial hyalinization and foci of squamous metaplasia of luminal epithelium in areas of inflammation. The connective tissue wall is fibrous with or without inflammatory infiltrate. SCC develops as a delayed complication occurring years after surgical intervention into the maxillary sinus especially after Le-fort I, II and III and midface osteotomies [20]. In the present case, history of previous surgery in the same site was non contributory.

Differentiation of GOC from mucosal cyst is rather difficult. The lining epithelium in mucosal cyst is of antral type, consisting of sero mucinous glands which of stains positive for alcian blue at pH 0.5. Also, since the mucosal cyst of antrum is a type of retention cyst which is non odontogenic, it seldom produces a palatal swelling as the growth occurs into the superior aspect of sinus from below. But GOC produces noticeable palatal swelling as cyst grow away from sinus inferiorly. In the present case, there was definite palatal swelling. Also clinical, radiographic, and histopathologic evidence along with special staining should be considered collectively in establishing a diagnosis.

In conclusion, the purpose of reporting this case was to add to the existing knowledge about this rare cyst which still has an uncertain nature.

 
   References Top

1.Padayachee A, Van Wyk CW (1987): two cystic lesions with features of both botryoid Odontogenic cyst and the central muco epidermoid tumor: Sialo odontogenic cyst? J Oral Pathol, 16:499-504.  Back to cited text no. 1    
2.Gardner DG; Kessler HP, Morency R, Schaffner DL (1988): The Glandular Odontogenic cyst: an apparent entity. J Oral Pathol 17; 359-66.  Back to cited text no. 2    
3.Sadeghi EM, Weldon LL, Kwon PH, Sampson E: Mucoepidermoid odontogenic cyst. Int J Oral Maxillofac Surg 20: 142-3.  Back to cited text no. 3    
4.High AS, Main DM, Khoo SP, PedlaR J, Hume WJ (1966): The polymorphous odontogenic cyst. J Oral Pathol Med 25:25-31.  Back to cited text no. 4    
5.Konstantinosl Tosios, Elina Kakarantza Angelopaulou, Nikiforos Kapranos(2000): Immunohistochemical study of bcl-2 protein, ki67 antigen and p53 protein in epithelium of glandular odontogenic cysts and dentigenous cysts. J Oral Pathol Med 29:139-44.  Back to cited text no. 5    
6.Rajendren R, Sivapathasundharam B (2005) Shafer, Hine, Levy's textbook of Oral Pathology 5th ed., Elsevier, India.  Back to cited text no. 6    
7.Koppang HS, Johannessen S, Haugen LK, Haanaes HR, Solheim T, Donath K (1998): Glandular odontogenic cyst (sialo-odontogenic cyst): report of two cases and literature review of 45 previously reported cases. J Oral Pathol Med 27:455-62.  Back to cited text no. 7    
8.Barreto, Luiz De Marco, DL Wagner HC, Gomez RS (2001): Glandular odontogenic cyst: absence of PTCH gene mutation. J Oral Pathol Med 30:125-8.  Back to cited text no. 8    
9.Shear M: Cysts of the oral region; (3rded.) Pg 71-74,1992,Varghese Publishing House, Mumbai.  Back to cited text no. 9    
10.Economopoulou P, Patrikiou A (1995): Glandular odontogenic cyst of the maxilla: report of a case. J Oral Maxillofac Surg, 53:834-7.  Back to cited text no. 10    
11.Kramer IRH, Pindborg JJ, Shear M (1992): Histological typing of odontogenic tumours, 2' Ed. Berlin : Springer-Verlag:38.  Back to cited text no. 11    
12.Shear M (1994): Developmental odontogenic cysts. An update. J Oral Pathol Med, 23:1-11.  Back to cited text no. 12    
13.Lucas's pathology of tumors of oral tissues (5`hed)pg: 136-138, Churchill Livingstone, London.  Back to cited text no. 13    
14.Patron M, Colmenero C, Laurrauri J (1991): Glandular odontogenic cyst: clinicopathologic analysis of three cases. Oral Surg Oral Med Oral Pathol 72: 71-4.  Back to cited text no. 14    
15.Ficarra Gy Chou L, Panzoni E (1990): Glandular odontogenic cyst (sialo-odontogenic cyst). A case report.JOral PatholMed19:331-3.  Back to cited text no. 15    
16.Ide F, Shimoyama T, Horie N (1996): Glandular odontogenic cyst with hyaline bodies: an unusual dentigerous presentation. J Oral Pathol Med 25:401-4.  Back to cited text no. 16    
17.Ramer M, Montazem A, Lane SL, Lumerman H (1997): Glandular odontogenic cyst. Report of a case and review of the literature. Oral Surg Oral Med Oral Pathol 84: 54-7.  Back to cited text no. 17    
18.Li TJ, Matthews JB (1994): Quantification of PCNA' cells within odontogenic jaw cyst epithelium. J Oral Pathol Med 23: 184-9.  Back to cited text no. 18    
19.Hussain K, Edmondson HD, Browne RM (1995): Glandular odontogenic cysts. Diagnosis and treatment. Oral Surg Oral Med Oral Pathol 79:593-602.  Back to cited text no. 19    
20.SugarAW and Walker DM (1990): J Oral Maxillofac Surg 28:264-267.  Back to cited text no. 20    


    Figures

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


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