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 Table of Contents  
CASE REPORT
Year : 2020  |  Volume : 32  |  Issue : 3  |  Page : 297-299

Dentigerous cyst with ameloblastomatous proliferation as well as calcifications: An unusual presentation


1 Department of Oral Pathology, Dr. R Ahmed Dental College and Hospital, Kolkata, West Bengal, India
2 Consultant Oral Radiologist, Nidaan Diagnostic Centre, Pune, Maharashtra, India

Date of Submission28-Dec-2019
Date of Decision06-Aug-2020
Date of Acceptance09-Aug-2020
Date of Web Publication29-Sep-2020

Correspondence Address:
Dr. Shreya S Saha
Bhalobasha Complex, Block No. B2, 10/1 R.K Das Rd, Haltu, Kolkata - 700078, West Bengal
India
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Source of Support: None, Conflict of Interest: None


DOI: 10.4103/jiaomr.jiaomr_209_19

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   Abstract 


Dentigerous cyst is an odontogenic cyst that is of developmental origin associated with the crown of an impacted tooth usually the third molars and canines. This is one of the most common types of developmental odontogenic cysts. The prognosis of dentigerous cyst is excellent but this cyst might undergo neoplastic transformation into ameloblastoma/squamous cell carcinoma/mucoepidermoid carcinoma. In this case report, we have studied the lining wall of the dentigerous cyst which has shown ameloblastomatous transformation along with sporadic calcification in the deeper layers of the connective capsule. This type of calcification rarely occurs with the dentigerous cyst.

Keywords: Ameloblastoma, calcification, connective tissue capsule, cyst lining, dentigerous cyst


How to cite this article:
Saha SS, Gandhoke HK, Mahato B, Deb T. Dentigerous cyst with ameloblastomatous proliferation as well as calcifications: An unusual presentation. J Indian Acad Oral Med Radiol 2020;32:297-9

How to cite this URL:
Saha SS, Gandhoke HK, Mahato B, Deb T. Dentigerous cyst with ameloblastomatous proliferation as well as calcifications: An unusual presentation. J Indian Acad Oral Med Radiol [serial online] 2020 [cited 2020 Oct 29];32:297-9. Available from: https://www.jiaomr.in/text.asp?2020/32/3/297/296579




   Introduction Top


Out of all odontogenic cysts of jaws, approximately 25% are dentigerous cysts.[1] The order of site of onset is regarded mostly in mandibular third molar followed by maxillary canines, mandibular premolars, maxillary third molars, and supernumerary teeth.[2] The male: female ratio was found to be 1.7:1.[3] Patients generally notice painless swelling. Histologically, dentigerous cyst consists of a fibrous wall and lined by non keratinized, stratified, squamous epithelium.[4] These cyst walls show a variety of epithelial appearances such as reduced enamel epithelium, transitional epithelium, and pseudoepitheliomatous hyperplasia.[5] The neoplastic transformation is highest in odontogenic keratocyst followed by the radicular cyst and dentigerous cyst.[6]


   Case Report Top


A 39-year-old male patient reported at the outpatient department of a Dental College and Hospital, Kolkata with a complaint of a painless swelling on the right side of the face. The swelling was asymptomatic for one year which gradually increased in size and suddenly he experienced pain for the last 10–15 days.

On clinical examination, solitary extraoral, well-demarcated swelling was present in the right premaxillary region obliterating the nasolabial fold.

Intraoral examination revealed retained upper right deciduous canine. A firm swelling measuring about 2cm × 1cm was present on concerned buccal gingiva and alveolus and extending from upper right lateral incisor till upper right first premolar. No tooth mobility was present.

A panoramic radiograph revealed a well-defined unilocular radiolucency extending from the periapical region of the right maxillary central incisor to the first premolar surrounding the crown in the neck region of mesially inclined unerupted right maxillary canine [Figure 1].
Figure 1: Orthopantomograph showing well-defined radiolucency around impacted right maxillary canine (preoperative)

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To evaluate further the extent of the lesion, cone beam computed tomography was advised. It showed the apex of impacted right maxillary canine was impinging the anterior border and floor of the maxillary sinus. Thinning and loss of buccal cortical plate and eggshell thinning in the lingual cortical plate was evident in the right maxillary central incisor region up to the concerned canine. External root resorption was present with the right maxillary central incisor, lateral incisor, and first premolar [Figure 2].
Figure 2: 3D reconstruction image showing expansion and destruction of bone around right maxillary canine region (preoperative)

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Based on clinical and radiographical findings, a provisional diagnosis of the dentigerous cyst was given. The patient underwent enucleation of the cystic lesion along with the extraction of affected teeth [Figure 3].
Figure 3: Post-operative picture after enucleation of the cystic lesion along with extraction of right maxillary impacted canine and right maxillary deciduous canine

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Histopathology revealed the presence of a cystic lining composed of odontogenic epithelium and stellate reticulum like cells were present in focal areas that were of variable thickness. In areas, the palisading arrangement of basal cells was seen. Some mucous cells were also interspersed within the epithelium. At one end in the deeper part of the capsule, basophilic calcifications of varying sizes were evident [Figure 4]. A final diagnosis of the dentigerous cyst with ameloblastomatous changes and calcification was given.
Figure 4: Histopathology showing ameloblastomatous changes of dentigerous cyst with basophilic calcifications

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   Discussion Top


On clinical examination, the finding of missing permanent teeth along with its retained deciduous tooth was suggestive of a dentigerous cyst which was asymptomatic.

Radiographically, the dentigerous cyst presents as a well-defined unilocular radiolucency, often with a sclerotic border. Since the epithelial lining is derived from the reduced enamel epithelium, this radiolucency typically and preferentially surrounds the crown of the tooth.

Similar findings have been noticed through orthopantomography and cone-beam computed tomography that is well defined unilocular expansile radiolucency with corticated margins.

Differential diagnosis included odontogenic cysts/tumors such as dentigerous cyst, odontogenic keratocyst, calcifying odontogenic cyst, adenomatoid odontogenic tumor, and ameloblastoma. Odontogenic keratocyst is intraosseous usually located in the periapical region and it can also cause root resorption. Calcifying odontogenic cyst sometimes involves an unerupted tooth especially canine and small radiopacities also noted in the radiograph. Dentigerous cyst is a unilocular cyst noted in relation to the crown of an impacted tooth. As the size of the cyst enlarges it can transform into ameloblastoma. Ameloblastoma may be unicystic or multicystic. Adenomatoid odontogenic tumors usually involve the crown of an unerupted tooth especially canine but it involves the tooth entirely.

  • In the present case, histopathology was consistent with dentigerous cysts with ameloblastomatous transformation. According to Vicker and Gorlin criteria, early ameloblastomatous changes include transformation to cuboidal or columnar basal cells with hyperchromatic nuclei, nuclear palisading with polarization, cytoplasmic vacuolization with intercellular spacing, and subepithelial hyalinization.[7] In 1933 Cahn first reported a case of ameloblastoma arising from a dentigerous cyst.[8]
  • Numerous immunohistochemical studies have been undertaken in the past few years, using a range of antibodies, to compare the cytokeratin contents of different odontogenic cysts. They proposed that their results suggested that two histogenic entities could occur that were indistinguishable by routine histological examination. They also pointed out that their results indicated that dentigerous, but not other cyst types, may share with some cases of ameloblastoma, the expression of cytokeratin polypeptide number 18.[9]
  • Our case report shows other unique histopathological findings that are basophilic globular calcifications present in the cystic capsule. These calcifications are cemento-osseous in nature. The development of such calcifications remains a mystery, although their origin is considered to be from the mesenchymal cells of the dental follicle. These cells can differentiate into cementoblasts or osteoblasts, producing a calcifying matrix.[10]


Normally dentigerous cyst never shows any kind of calcifications as searched in the literature; also, the presence of ameloblastomatous changes is very rarely seen. Our present case report in dentigerous cyst shows ameloblastomatous changes as well as calcifications which makes it very unique.


   Conclusion Top


It is very important to determine whether a dentigerous cyst is undergoing any ameloblastomatous changes. If ameloblastomatous changes are evident, the treatment option includes enucleation along with curettage which is essential to prevent its recurrence. Dentigerous cyst shows the potential for neoplastic transformation to ameloblastoma, mucoepidermoid carcinoma, squamous cell carcinoma, adenomatoid odontogenic tumor, and odontome. The whole cyst should be actively evaluated to rule out any such transformation.

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.
Umredkar AB, Gaur P, Anand A, Ganvir S, Ansari M. Dentigerous cyst: An unusual presentation with angiomatous changes. J Med Sci Health 2017;3:39-42.  Back to cited text no. 1
    
2.
Manjunatha BS, Chikkaramaiah S, Panja P, Koratagere N. Impacted maxillary second premolars: A report of four cases.BMJ Case Rep 2014;2014:bcr2014205206.doi: 10.1136/bcr-2014-205206.  Back to cited text no. 2
    
3.
Kilinc A, Gundogdu B, Saruhan N, Yalcin E, Ertas U, Urvasizoglu G. Odontogenic and nonodontogenic cysts: An analysis of 526 cases in Turkey. Niger J Clin Pract 2017;20:879-83.  Back to cited text no. 3
[PUBMED]  [Full text]  
4.
Kalaskar RR, Tiku A, Damle SG. Dentigerous cyst of anterior maxilla in a young child-A case report. J Indian Soc Pedod Prev Dent 2007;25:187-90.  Back to cited text no. 4
[PUBMED]  [Full text]  
5.
Huang G, Moore L, Logan RM, Gue S. Histological analysis of 41 dentigerous cysts in a paediatric population. J Oral Pathol Med 2019;48:74-8.  Back to cited text no. 5
    
6.
Ali A, Asif M, Ahmad B, Jamal S, Ali I, Khadim MT. Stromal expression of CD10 by immunohistochemistry in odontogenic keratocyst (OKC), dentigerous and radicular cysts and its correlation with local recurrence and aggressive behaviour. Asian Pac J Cancer Prev 2019;20:249-53.  Back to cited text no. 6
    
7.
Vickers RA, Gorlin RJ. Ameloblastoma: Delineation of early histopathologic features of neoplasia. Cancer 1970;26:699-710.  Back to cited text no. 7
    
8.
Cahn LR. The dentigerous cyst as a potential adamantinoma. Dent Cosmos1993;75:889-93.  Back to cited text no. 8
    
9.
Shear M, Speight P. Dentigerous cyst. In: Shear M, Speight P, editors. Cysts of the Oral and Maxillofacial Regions. 4th ed. Copenhagen, Denmark: Blackwell Munksgaard; 2007. p. 74-5.  Back to cited text no. 9
    
10.
Cho YA, Yoon HJ, Hong SP, Lee JI, Hong SD. Multiple calcifying hyperplastic dental follicles: Comparison with hyperplastic dental follicles. J Oral Pathol Med 2011;40:243-9.  Back to cited text no. 10
    


    Figures

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



 

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