Contemporary Clinical Dentistry
   
  Home | About us | Editorial board | Search
Ahead of print | Current Issue | Archives | Advertise
Instructions | Online submission| Contact us | Subscribe |

 

Login  | Users Online: 288  Print this pageEmail this pageSmall font sizeDefault font sizeIncrease font size 



 
 Table of Contents  
CASE REPORT
Year : 2013  |  Volume : 4  |  Issue : 1  |  Page : 108-111  

Odonto calcifying cyst


1 Department of Oral Medicine and Radiology, Sree Balaji Dental College and Hospital, Chennai, Tamil Nadu, India
2 Department of Oral Pathology, Sree Balaji Dental College and Hospital, Chennai, Tamil Nadu, India

Date of Web Publication9-May-2013

Correspondence Address:
Nalini Aswath
Department of Oral Medicine and Radiology, Sree Balaji Dental College and Hospital, Chennai 600 100, Tamil Nadu
India
Login to access the Email id

Source of Support: None, Conflict of Interest: None


DOI: 10.4103/0976-237X.111629

Rights and Permissions
   Abstract 

The calcifying odontogenic cyst (COC) is reported to be associated with odontoma in 24% of cases. Separation of the cases of calcifying odontogenic cyst associated with odontoma (COCaO) may lead to a better understanding of the pathogenesis of this lesion. The literature revealed 52 cases of COCaO. The male to female ratio was 1:1.9, with a mean age of 16 years. Most common location was the maxilla (61.5%). The radiographic appearance of most cases (80.5%) was a well-defined, mixed radiolucent-radiopaque lesion. Histologically, the lesions consisted of a single large cyst with tooth-like structures as an integral part, giving the impression of a single lesion. In addition to the unique histologic features, differences in gender and distribution were found between the cases of COCaO and those of simple COC. COCaO may be regarded as a separate entity and classified as a benign, mixed odontogenic tumor. The term odontocalcifying odontogenic cyst is suggested.

Keywords: Odontogenic cyst, calcification, odontome


How to cite this article:
Aswath N, Mastan K, Manikandan T, Samuel G. Odonto calcifying cyst. Contemp Clin Dent 2013;4:108-11

How to cite this URL:
Aswath N, Mastan K, Manikandan T, Samuel G. Odonto calcifying cyst. Contemp Clin Dent [serial online] 2013 [cited 2019 Dec 7];4:108-11. Available from: http://www.contempclindent.org/text.asp?2013/4/1/108/111629


   Introduction Top


The calcifying odontogenic cyst (COC) was first defined as a separate entity by Gorlin et al., in 1962. [1] The COC represents about 1% of jaw cysts, and although it may occur in soft tissue, it is most commonly found within bone. [2],[3] Both the intraosseus and extra osseous forms occur with about equal frequency in the maxilla and mandible, mainly in the incisor and canine areas. [4],[5],[6],[7] Although, the peripheral COC affects individuals in the sixth decade of life, the central variant is observed most commonly in the second decade and does not show gender predilection.

COC is usually asymptomatic and may be an incidental radiographic finding. [2] Radiographically, the lesion appears as a unilocular or multilocular well-defined radiolucency that may contain small irregular calcified bodies of varying sizes, and it may be associated with an odontoma or an unerupted tooth. [2],[8],[9],[10]

Ghost cells are a characteristic feature of COC, although they also occur (less frequently) in other lesions such as the odontoma. In addition, dentin like material and enamel may be present in the COC. The World Health Organization (WHO)'s International Classification of Odontogenic Tumors and Allied Lesions [11] define it as "a non-neoplastic lesion" and mention the possible association with other odontogenic lesions, such as odontoma. Nevertheless, few cases of true odontoma found concurrently with COC have been reported. [12],[13],[14]


   Case Report Top


A 13-year-old boy reported with the chief complaints of space in the maxillary front tooth region since 1 month [Figure 1].
Figure 1: A 13-year-old boy complaints of space in the maxillary front tooth region

Click here to view


History reveals that the patient visited a dentist for improper arrangement of the maxillary front tooth region and had undergone uneventful extraction. On clinical examination, extra orally there was no obvious facial asymmetry.

Intra oral examination showed an ill-defined swelling of the buccal gingiva in the region of right maxillary central and lateral incisors measuring 1.5 cm × 1 cm in size. The swelling was slightly obliterating the labial vestibule; surface over the swelling was smooth, normal in color and texture. On palpation, the swelling was bony hard in consistency and non-tender [Figure 2].

On correlating the patient's complaint, with the history and clinical examination a provisional diagnosis of benign tumor of maxilla was made.
Figure 2: Intra oral examination showing an ill-defined swelling of the buccal gingiva

Click here to view


On radiographic examination, IOPA Intra Oral Periapical Radiograph showed radiopaque mass with multiple teeth like structures present in relation to 11, 12, 13 measuring 0.5 cm in size. Occlusal radiograph showed expansion and thinning of the labial cortical plate [Figure 3]. OPG Ortho Pan Tomography shows a tooth like radio opaque mass in relation to right central and lateral incisor surrounded by a well-defined radiolucent rim towards the inferio-left lateral aspect of the mass [Figure 4].
Figure 3: Occlusal radiograph showed radiopaque mass with multiple teeth like structures present in relation to 11, 12, 13 with expansion and thinning of the labial cortical plate

Click here to view
Figure 4: OPG Ortho Pan Tomograph showing a lesion in relation to the right central and lateral incisor

Click here to view


The radiographic differential diagnosis that were considered are odontome, cystic odontoma, multiple supernumerary teeth, COC, Adenomatoid odontogenic tumour, ameloblastic fibro-odontoma.

The patient was admitted, and enucleation of the lesion was done under local anesthesia. The lesion was easily detached from the surrounding bone and was removed together with hard tissue. The excised specimen consisted of a cystic sac approximately 2 cm in diameter; the odontoma-like hard tissue was attached to the cystic wall.

Microscopically, the histopathological section of the specimen showed odontogenic lining epithelium which was single layered in one area and two to three layers in the remaining areas lyning the cystic cavity [Figure 5]. Above the odontogenic epithelium stellate reticulum like cells were arranged. Eosinophilic masses suggestive of ghost cells were seen. A fibrous connective tissue wall was also present [Figure 6].
Figure 5: Odontogenic epithelium lining the cystic cavity

Click here to view
Figure 6: Stellate reticulum like cells arranged above the odontogenic epithelium, the eosinophilic masses suggestive of ghost cells

Click here to view


The decalcified section of the hard tissue showed enamel and dentin like material arranged in disorganized fashion [Figure 7]. The histopathologic features were suggestive of COC associated with complex composite odontoma.
Figure 7: Decalcified section showing hard tissue with enamel and dentin like material arranged in disorganized fashion

Click here to view



   Discussion Top


Since 1971, COC has been described by the WHO as a non-neoplastic cystic lesion in which the epithelial lining shows a well-defined basal layer that is often many cells thick and that may resemble the stellate reticulum of an enamel organ, and masses of ghost epithelial cells that may be in the epithelial cyst lining or in the fibrous capsule. [11] The ghost cells may become calcified and dysplastic dentin may be laid down next to the basal cell layer of the epithelium. It may be associated with complex odontoma or with tissue resembling an ameloblastic fibro-odontoma.

Praetorius [8] proposed a sub-classification for the heterogeneous group of COC's, in which the cystic lesions were separated from the neoplasms (solid lesions). These researchers further divided the cystic entity into three types, the simple unicystic type, the unicystic odontoma-producing type, and the ameloblastomatous proliferating type. Other authors have reported only 13-21% of the cysts to be peripheral (extraosseous) lesions. [6],[7] The area affected in this case has been considered the most commonly affected site: About 65% of the reported cases were found in the incisor-canine area. [8] The radiographic findings of this case (unilocular radiolucency with a radiopaque mass and well-circumscribed borders) are encountered in the majority of odontoma-producing intraosseous COC.

The radiopaque mass observed in the panoramic radiograph of this case, later diagnosed as odontoma components, are considered to be present in 24-50% of the reported cases. [7] These cases were classified as a subtype of COC under the term odontoma-producing type, or as a cystic variant associated with odontoma. [11] Hirschberg et al, [15] proposed that this variant to be classified as a separate entity as odontocalcifying odontogenic cyst. They observed that this variant was more prevalent in females, with a mean age at discovery of 16 years (compared with 34.4 years in the simple COC group) and that most cases were located in the maxilla. The findings of our case were mostly in accordance with the above mentioned observations. This variant may be more common in younger adults, but it may also be discovered in older persons. The possibility of occurrence of an odontoma associated with COC may result from factors present at a certain age, but it seems that the same odontogenic epithelium is present in COC and in this particular variant. Furthermore, it seems that the same characteristic epithelium is involved in the various clinical presentations of this lesion.

Finally, most authorities regard the COC as a lesion with little, if any, tendency to recur, and enucleation is the most commonly reported method of treatment. Several cases of recurrent COC have been reported. Wright et al, [16] also have reported that recurrence has developed 5 years or more following initial therapy. McGowan and Browne [17] stated that follow-up periods of up to 10 years are advisable. Therefore, further follow-up examination will also be conducted in the present case, considering the neoplastic nature of the lesion.


   Summary Top


A COC associated with a complex odontoma is described. The presence of reduced enamel epithelium showing extensive proliferation accompanied by dentinoid and enameloid formation, and the morphologic connection of the lining epithelium of the cyst with the enamel epithehum, indicate that the possible origin of the cyst is the enamel epithelium of the odontoma.

 
   References Top

1.Gorlin RJ, Pindborg JJ, Odont, Clausen FP, Vickers RA. The calcifying odontogenic cyst: A possible analogue of the cutaneous calcifying epithelioma of Malherbe. An analysis of fifteen cases. Oral Surg Oral Med Oral Pathol 1962;15:1235-43.  Back to cited text no. 1
    
2.Odell EW, Morgan PR. Biopsy Pathology of the Oral Tissues. London, UK: Chapman and Hall Medical; 1998. p. 229-33.  Back to cited text no. 2
    
3.Shamaskin RG, Svirsky JA, Kaugards GE. Intraosseous and extraosseous calcifying odontogenic cyst (Gorlin Cyst). J Oral Maxillofac Surg 1989;47:562-5.  Back to cited text no. 3
    
4.Altini M, Ferman AG. The calcifying odontogenic cyst: Eight new cases and a review of the literature. Oral Surg Oral Pathol Oral Radiol 1975;40:751-9.  Back to cited text no. 4
    
5.Freedman PD, Lumerman H, Gee JK. Calcifying odontogenic cyst: A review and analysis of seventy cases. Oral Surg Oral Med Oral Pathol 1975;40:93-106.  Back to cited text no. 5
    
6.Nagao T, Nakajima T, Fukushima M, Ishiki T. Calcifying odontogenic cyst: A survey of 23 cases in the Japanese literature. J Maxillofac Surg 1983;11:174-9.  Back to cited text no. 6
    
7.Buchner A. The central (intraosseous) calcifying odontogenic cyst: An analysis of 215 cases. J Oral Maxillofac Surg 1991;49:330-9.  Back to cited text no. 7
    
8.Buchner A, Merrell PW, Carpenter WM, Leider AS. Central (intraosseous) calcifying odontogenic cyst. Int J Oral Maxillofac Surg 1990;19:260-2.  Back to cited text no. 8
    
9.Praetorius F, Hjorting-Hansen E, Gorlin RI, Vickers RA. Calcifying odontogenic cyst: Range, variations and neoplastic potential. Acta Odontol Scand 1981;39:227-40.  Back to cited text no. 9
    
10.Progrel MA. Treatment of keratocysts: The case for decompression and marsupialization. J Oral Maxillofac Surg 2005;63:1667-73.  Back to cited text no. 10
    
11.Pindborg JJ, Kramer IRH, Torloni H. Histological typing of odontogenic tumors, jaw cyst and allied lesions. Geneva: World Health Organization; 1971.  Back to cited text no. 11
    
12.Nagao T, Nakajima T, Fukushima M, Ishiki T. Calcifying odontogenic cyst: a survey of 23 cases in the Japanese literature. J Maxillofac Surg 1983;11:174-9.  Back to cited text no. 12
    
13.Eda S, Kawahara H. Yamamura T. Imaizumi I, Ohi M. A case of calcifying odontogenic cyst associated with odontoma. Bull Tokyo Dent Coll 1971;12:1-4.  Back to cited text no. 13
    
14.Johnson A 3 rd , Fletcher M, Gold L, Chen SY. Calcifying odontogenic cyst: A clinicopathologic study of 57 cases with immunohistochemical evaluation for cytokeratin. J Oral Maxillofac Surg 1997;55:679-83.  Back to cited text no. 14
    
15.Hirshberg A, Kaplan I, Buchner A: Calcifying odontogenic cyst associated with odontoma: A possible separated entity (Odontocalcifying odontogenic cyst). J Oral Maxillofac Surg 1994; 52:555,  Back to cited text no. 15
    
16.Wright BA, Bhardwaj AK, Murphy D: Recurrent calcifying odontogenic cyst. Oral Surg Oral Med Oral Path 1984;58:579-83.  Back to cited text no. 16
    
17.McGowan RH, Browne RM: The calcifying odontogenic cyst: A problem of preoperative diagnosis. Br J Oral Surg. 1982;20:203  Back to cited text no. 17
    


    Figures

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


This article has been cited by
1 Two unique cases of CCOT in the maxillary posterior region
Jira Chindasombatjaroen,Sopee Poomsawat,Kiatanant Boonsiriseth
Oral Surgery, Oral Medicine, Oral Pathology and Oral Radiology. 2014;
[Pubmed] | [DOI]



 

Top
 
  Search
 
    Similar in PUBMED
   Search Pubmed for
   Search in Google Scholar for
 Related articles
    Access Statistics
    Add to My List *
* Registration required (free)  

 
  In this article
    Abstract
   Introduction
   Case Report
   Discussion
   Summary
    References
    Article Figures

 Article Access Statistics
    Viewed1856    
    Printed44    
    Emailed0    
    PDF Downloaded180    
    Comments [Add]    
    Cited by others 1    

Recommend this journal