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 Table of Contents  
CASE REPORT
Year : 2020  |  Volume : 7  |  Issue : 2  |  Page : 117-119

Maxillary calcifying epithelial odontogenic tumor


Department of Oral Pathology and Microbiology, Annoor Dental College, Ernakulam, Kerala, India

Date of Submission21-Mar-2019
Date of Decision24-Oct-2019
Date of Acceptance17-Mar-2020
Date of Web Publication21-May-2020

Correspondence Address:
Dr. P R Bindhu
Department of Oral Pathology and Microbiology, Annoor Dental College, Muvattupuzha, Ernakulam, Kerala
India
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Source of Support: None, Conflict of Interest: None


DOI: 10.4103/sjos.SJOralSci_27_19

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  Abstract 


Calcifying epithelial odontogenic tumor (CEOT) is a rare benign epithelial odontogenic tumor, having characteristic clinical and histopathological features. CEOT usually affects middle-aged adults without any gender predilection. Most of these cases are intraosseous and appear predominantly in the mandibular posterior region. It is not commonly found in the maxillary region; however, if appear, it can easily involve the surrounding areas including the maxillary sinus. Here, we discuss a case of CEOT in the maxillary premolar–molar region, the common site being the posterior mandible.

Keywords: Benign, characteristic, maxillary, odontogenic tumor, posterior


How to cite this article:
Bindhu P R, Krishnapillai R, Thomas P. Maxillary calcifying epithelial odontogenic tumor. Saudi J Oral Sci 2020;7:117-9

How to cite this URL:
Bindhu P R, Krishnapillai R, Thomas P. Maxillary calcifying epithelial odontogenic tumor. Saudi J Oral Sci [serial online] 2020 [cited 2020 Sep 22];7:117-9. Available from: http://www.saudijos.org/text.asp?2020/7/2/117/284658




  Introduction Top


Calcifying epithelial odontogenic tumor (CEOT), reported as a distinct entity by the Danish pathologist JJ Pindborg in 1955, is a rare benign odontogenic neoplasm constituting around 0.4%–3% of all intraosseous tumors.[1],[2] The name “Pindborg tumor” was suggested as a mark of honor for this pathologist.[2],[3] It is a slow-growing, painless neoplasm with a locally aggressive nature, common in the fourth and fifth decades of life without any gender predilection.[1] This neoplasm appears as an intraosseous mass of the mandible associated with an impacted tooth without any gender preference.[4] Here, we report a case of CEOT in a middle-aged female.


  Case Report Top


A 42-year-old female reported to the department of oral medicine and radiology with a gradually increasing swelling on the right upper jaw for 10 months. The lesion started as a small enlargement and gradually increased to the present size. It was painless, asymptomatic, and without any pus discharge. There was no extraoral swelling. Intraorally, the lesion was firm to hard in consistency and oval in shape extending from the maxillary first premolar to second molar, measuring 5 cm × 3 cm. Overlying mucosa appeared normal. X-ray and computed tomography revealed a radiolucent lesion with flecks of radiopacities associated with impacted maxillary second and third molars [Figure 1]a, [Figure 1]b, [Figure 1]c.
Figure 1: (a) Computed tomography, (b) scanning electron microscopy, (c) orthopantomogram showing a radiolucent lesion with scattered radiopacities and cortical plate expansion. (d) Gross specimen: Multiple bits of lesional tissue and associated teeth

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The lesion was surgically excised with a margin of normal healthy bone and sent to the oral pathology department. The gross specimen was composed of numerous small soft tissue bits with associated teeth [Figure 1]d. Hematoxylin and eosin-stained sections revealed sheets of polyhedral cells with eosinophilic cytoplasm and prominent intercellular bridges. There is also evidence of amyloid-like material both intra- and extra-cellularly and areas of calcifications [Figure 2]. Based on histopathology, a diagnosis of calcifying epithelial odontogenic cyst was given.
Figure 2: Photomicrograph showing (a) amyloid-like material, areas of hemorrhage, and calcification (H and E, ×4), (b and c) polygonal squamous cells with extracellular amyloid-like material and calcification (H and E, ×40), (d) clear cells

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Once the surgical wound healed, the patient was referred to the department of prosthodontics. Maxillary impression was taken using irreversible hydrocolloid impression material, casted with Type III dental stone and prosthesis fabricated using heat cure acrylic resin. She was advised review appointments every 6 months. After the first two appointments, the patient was perfectly normal without any evidence of recurrence.


  Discussion Top


CEOT named “Pindborg tumor” is a rare benign neoplasm making up <1% of all odontogenic tumors.[2] CEOT commonly present as a central lesion of the posterior ramus of the mandible (85%).[5] Pindborg tumor exists in two forms: intraosseous (94%) and extraosseous (6%).[6] This tumor was also known by the names adenoid ameloblastoma, unusual ameloblastoma, and cystic odontoma. The tumor is considered to be originating from dental lamina remnants, stratum intermedium, reduced enamel epithelium, or oral epithelium.[7] Many researchers support its origin from the stratum intermedium layer of the enamel organ due to morphologic resemblance of the tumor cells to normal stratum intermedium cells and a high alkaline phosphatase and adenosine triphosphate activity.[8]

Although the disease tends to occur over a wide age range, it is predominantly seen in the third to sixth decades of life with an equal sex predilection. Peripheral CEOT commonly exhibits a predilection to anterior gingiva and presents as a nodular swelling. Literature review reveals that it appears predominantly in the mandible compared to the maxilla (2:1). Most of the cases appear in the premolar molar region, and more than 50% of cases occur associated with an impacted/unerupted tooth, especially the mandibular third molar.[9] Contrary to that, our case was located in the posterior maxilla.[10]

The disease tends to be asymptomatic, often discovered during routine radiography, but may occasionally present as a painless, slow-growing, expansile intrabony swelling with thinning of the cortical plates or even perforation.[8] Maxillary lesions tend to be more aggressive as it can invade the maxillary sinus or ethmoid sinus causing pain, nasal obstruction, epistaxis, and head ache. Hence, early diagnosis of maxillary CEOT is an important factor with regard to prognosis. Radiographic appearance of this lesion is highly variable demonstrating an irregular radiolucency (unilocular/multilocular) or a mixed radiolucent radiopaque lesion.

The tumor is composed of sheets, islands, and strands of polyhedral epithelial cells, within a fibrous connective tissue stroma. Large areas of amorphous eosinophilic amyloid-like extracelluar material and concentric rings of calcifications (Liesegang ring) are the characteristic features of this neoplasm.[11],[12] Variants of CEOT include clear cell, myoepithelial cell, pigmented, Langerhans cell containing, bone and cementum forming, and cystic and noncalcifying. Hybrid tumors have also been reported in the literature (CEOT and adenomatoid odontogenic tumor, CEOT, and ameloblastoma).[10]

Conservative surgical resection with a margin of normal surrounding bone is the treatment of choice. The recurrence rate of this tumor has been reported to be 10%–15%.[13] Overall prognosis seems to be good, though there is a report of malignant transformation and metastasis.[12] Prosthetic rehabilitation is advised based on the degree of bone loss. Our case also was excised conservatively with a margin of apparently unaffected normal bone. Follow-up 6 months and 1 year after surgery revealed no recurrence.


  Conclusion Top


From its first description in 1955, the number of reported cases of CEOT in the literature has increased to over 200 cases at present. We indent to present this case as maxilla is a rare location for this tumor to occur. Clinical, radiographical, and histopathological aspects have to be considered in the diagnosis of CEOT. Maxillary cases require more careful treatment planning and execution, as it can encroach upon the adjacent structures. We also would like to emphasize that total removal of the neoplasm is an absolute necessity in the prevention of 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.
Srikrishna K, Vinod VC, Venkateswarlu M, Sreedevi K, Prasad Reddy GS. Pindborg tumor: Review of literature and case reports. JIAOMR 2011;23:660-3.  Back to cited text no. 1
    
2.
Harshavardhan T, Naag S, Umamagesh DP, Priyadarshini E. Maxillary calcifying epithelial odontogenic tumor presenting at a postextraction site: A case report and brief review of literature. J Indian Acad Oral Med Radiol 2017;29:333-6.  Back to cited text no. 2
  [Full text]  
3.
Malik SN, Alam MK, Shahina M, Siddique S, Prabhu VD. Calcifying epithelial odontogenic tumor (CEOT). Bangladesh J Med Sci 2014;13:14-9.  Back to cited text no. 3
    
4.
Pereira OH, de Carvalho LP, Brasileiro Junior VL, de Figueiredo CR. Calcifying epithelial odontogenic tumor. Case Rep Pathol 2013;Article ID 725380:3.  Back to cited text no. 4
    
5.
Kaushal S, Mathur SR, Vijay M, Rustagi A. Calcifying epithelial odontogenic tumor (Pindborg tumor) without calcification: A rare entity. J Oral Maxillofac Pathol 2012;16:110-2.  Back to cited text no. 5
  [Full text]  
6.
Yanik S, Ayranci F, Bozdag Z, Oznalcın O, Aras MH. A rare case of multilocular calcalcifying epithelial odontogenic tumor. Int J Clin Exp Med 2016;9:17078-81.  Back to cited text no. 6
    
7.
More CB, Vijayvargiya R. Intraosseous calcifying epithelial odontogenic (Pindborg) tumor: A rare entity. J Oral Maxillofac Pathol 2015;19:269.  Back to cited text no. 7
[PUBMED]  [Full text]  
8.
Singh N, Sahai S, Singh S, Singh S. Calcifying epithelial odontogenic tumor (Pindborg tumor). Natl J Maxillofac Surg 2011;2:225-7.  Back to cited text no. 8
[PUBMED]  [Full text]  
9.
Angadi PV, Rekha K. Calcifying epithelial odontogenic tumor (Pindborg tumor). Head Neck Pathol 2011;5:137-9.  Back to cited text no. 9
    
10.
Narayanaswamy SA, Rao K, Dutt CS, Smitha T. Cystic variant of calcifying epithelial odontogenic tumor. IJOCR 2017;5:345-7.  Back to cited text no. 10
    
11.
Yanduri S, Kumar BV. Liesegang rings: The misnomer in calcifying epithelial odontogenic tumor? J Adv Clin Res Insights 2016;3:224-5.  Back to cited text no. 11
    
12.
Neville B, Damm D, Allen C, Bouquot J. Oral and Maxillofacial Pathology. 3rd ed. Philadelphia: W.B. Saunders; 2008.  Back to cited text no. 12
    
13.
Akhtar K, Khan N, Zaheer S, Sherwani R, Hasan A. Pindborg tumor in an adolescent. Oman Med J 2010;25:47-8.  Back to cited text no. 13
    


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