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CASE REPORT
Year : 2019  |  Volume : 7  |  Issue : 2  |  Page : 44-48

Unusual location of calcifying epithelial odontogenic tumor


Department of Oral and Maxillofacial Surgery, Istanbul University Faculty of Dentistry, Istanbul, Turkey

Date of Web Publication1-Oct-2019

Correspondence Address:
Aydın Gümüşdal
Küçükyali Mah. Mektep Cad., Yavru Sok., No: 10 D: 9 Maltepe, Istanbul
Turkey
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Source of Support: None, Conflict of Interest: None


DOI: 10.4103/jomr.jomr_18_19

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  Abstract 


Calcifying epithelial odontogenic tumor (CEOT), also known as Pindborg tumor, is locally aggressive and rare odontogenic epithelial neoplasm. It is mostly located in the posterior mandible. Maxillary occurrence is extremely rare, and this location leads the lesion to become more aggressive and rapidly involves to the surrounding tissues. Treatment of the lesion has a variety of choice due to location of the lesion, rapid invasion, proximity to vital structures, and early diagnosis. We present clinical, radiographic, and histopathologic examinations and surgical operation of a CEOT in the alveolar process between maxillary first and second molar roots.

Keywords: Aggressive, calcifying epithelial odontogenic tumor, odontogenic tumor, Pindborg, treatment


How to cite this article:
Cankaya B, İşler SC, Gümüşdal A, Genç B, Asadov C. Unusual location of calcifying epithelial odontogenic tumor. J Oral Maxillofac Radiol 2019;7:44-8

How to cite this URL:
Cankaya B, İşler SC, Gümüşdal A, Genç B, Asadov C. Unusual location of calcifying epithelial odontogenic tumor. J Oral Maxillofac Radiol [serial online] 2019 [cited 2019 Dec 7];7:44-8. Available from: http://www.joomr.org/text.asp?2019/7/2/44/268238




  Introduction Top


Calcifying epithelial odontogenic tumor (CEOT) is a rare benign, locally aggressive, odontogenic lesion founded by Pindborg in 1955.[1] It constitutes 1% of all odontogenic tumors presenting intraosseous-extraosseous variants.[2],[3] Most of the cases are intraosseous that show up unilocular or multilocular radiolucencies.[4] Clinically, CEOTs are usually painless and develop slowly except for maxillary lesions. CEOT has unique histopathological features for its diagnosis. These are sheets, strands, and discrete islands of polyhedral epithelial cells supported by a fibrous connective tissue stroma.[5] CEOT usually locates in the premolar and molar regions of the mandible and often shows itself as a slowly growing painless mass.[6] The origin of this neoplasm is not certain of which can come from the oral epithelium, reduced enamel epithelium, stratum intermedium, or dental lamina remnants.[7] The differential diagnosis must be made with adenomatoid odontogenic tumor, calcifying odontogenic cyst, ameloblastic fibro-odontoma, and odontoma.[8] In our case, there is a rapid growing, expansive, and painless lesion that has an unilocular, demarcated, and homogeneous radiological appearance with too small calcified points between the first and second molar roots at the left maxilla.


  Case Report Top


A systemically healthy 39-year-old female patient was applied to our clinic with an asymmetry of the left maxillary molar region. According to her medical history, amorphous development started 2 months ago. The lesion was showed semi-solid stiffness in the palpation examination. The gingival mucosa was appeared in natural view except swelling due to expansion of the cortical bone. No luxation of teeth that associated with the lesion is seen. According to radiological examination, there was a unilocular, demarcated, and homogeneous radiolucency between the first and second molar teeth roots [Figure 1] and [Figure 2]. Lamina dura had erased around the distal root of the tooth, number 26. No calcified mass was detected in panoramic radiograph [Figure 3]. Small calcified points were seen only in computed tomography (CT) [Figure 4], [Figure 5], [Figure 6]. According to clinical and radiological examinations and medical history, we primarily planned biopsy operation. Histopathological examination revealed that the lesion was CEOT. The results of the histopathological examination were tumor spots in the connective tissue which is rich in collagen fibers. Eosinophil, amyloid, or transparent odontogenic epithelium cell groups are seen. According to the results of the biopsy, we decided to remove the lesion. During surgical procedure, after local anesthesia and triangular flap lifting, we primarily extract the teeth, numbers 26 and 27 [Figure 7], [Figure 8], [Figure 9], [Figure 10]. After that, we detected the capsule-like structure of the lesion, and it was allowed us to enucleate the lesion easily. Considering the possibility of recurrence, the surrounding bone has been corrected by bur. Operation area was closed with 3.0 silk sutures after debridement with saline solution. The definitive histopathological result is CEOT. End of the postoperative 2 months, no discernible complications and recurrence were seen [Figure 11], [Figure 12], [Figure 13], [Figure 14], [Figure 15], [Figure 16].
Figure 1: Sagittal section of cone-beam computed tomography view of the lesion

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Figure 2: Coronal section of cone-beam computed tomography view of the lesion

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Figure 3: The initial panoramic radiograph of the patient with the lesion at the left maxilla. Calcified focal spot is not seen in the panoramic radiograph

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Figure 4: Axial section of cone-beam computed tomography view of the lesion. Bookmarked area is a calcified focal spot

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Figure 5: Sagittal section of cone-beam computed tomography view of the calcified focal spot

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Figure 6: Coronal cone-beam computed tomography view of the calcified focal spot

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Figure 7: (a) Preoperative, intraoral view and (b) preoperative, intraoral view

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Figure 8: Triangular flap is removed to reach the lesion, and the teeth, numbers 26 and 27, are extracted

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Figure 9: The removed lesion and the extracted teeth

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Figure 10: Postoperative 1-year follow-up

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Figure 11: Six-month follow-up

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Figure 12: Three-year follow-up

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Figure 13: Three-year follow-up, axial section of cone-beam computed tomography view

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Figure 14: Three-year follow-up, sagittal section of cone-beam computed tomography view

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Figure 15: Three-year follow-up, coronal section of cone-beam computed tomography view

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Figure 16: Post-operative 1 year follow-up

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  Discussion Top


The literature reports that CEOT (Pindborg tumor) usually evinces as painless intumescences that cause a slow bone expansion. CEOT is a rare odontogenic neoplasm that defines about 1% of all odontogenic tumors.[9],[10] It has been proposed that CEOT stems from the stratum intermedium of the dental organ or stems from the dental lamina. CEOT can show up at any age; however, it is mostly seen between 30 and 50 ages, with no sexual discriminations.[11] The wide majority of cases consist in the posterior mandible. Rare samples have also been notified with the anterior gingiva being the densest location.[12],[13] In addition to this maxillary tumors might be more aggressive and cause syptoms like stuffiness and epstaxis in the nasal cavity.[12] There is a broad variation in clinical, radiological, and histological views.

Radiographically, CEOTs seem as unilocular or multilocular radiolucencies with or without radiopacities. Almost 50% of the cases are corporate with unerupted teeth and seem radiographically as dentigerous cysts. The central type of CEOT is usually located in the premolar and molar area, with a mandibular to maxillary proportion of 2:1 or 3:1.[14] In some cases, particularly in tumors of relatively short period, the calcification is very small and may not be defined on radiographs.[15] When an unerupted tooth is incorporated with the tumor, the radiopacity seems located near to tooth crown.[16]

Histologically, many samples indicate solid proliferation of polyhedral epithelial cells composed in distinct islands, strands, or sheets in a background of fibrous stroma with changing quantity of amorphous, eosinophilic, and amyloid-like material and calcifications.[17] Other variants of the classic semblance have been known and contain tumors with minimal amyloid-like material and calcification, prominent amyloid-like material and minimal epithelial cells, and a mainly clear-cell variant of CEOT.[18] However, it should be explained that the diagnosis of a true malignant CEOT is usually made with stimulation, as CEOT often represents important hyperchromatism and pleomorphism, but CEOT is a benign tumor whose biological attitude was once believed to be similar to that of ameloblastoma.[18] It is largely defined as a slowly growing mass, occasionally associated with a long history of evolution. However, in most cases, there are little data on tumor term, and for this reason, the true growth rate of the tumor is uncertain.[19]

The assessment of this case can be considered relatively short with rapid growth of tumor mass. The treatment for CEOT has ranged from basic enucleation or curettage to radical and wide resection such as hemimandibulectomy or hemimaxillectomy. Surgical procedure differs from lesion to lesion because of the radiological and histological varieties of the lesions. The lesion usually has benign character; even it has a high recurrence rate, malignancy is extremely rare. On the other hand, postoperative follow-ups should continue at least for 5 years.[20]

Maxillary located, rapid developing CEOT caused an expansion and loss of the lamina dura in the distal section of of the tooth number 26. Calcified area covers small portion of the lesion. Encapsulated structure, easily detachment from the bone, led us to enucleation the lesion. Postoperative follow-ups are still continuing.

CEOT's location apart from literature is usually in mandible. CEOT's location is between the roots of the teeth and periphery of the impacted teeth, therefore it can be confused with dentigerous cyst. If there is no capsule, curettage should be done. If there is a capsule, enucleation should be done. There is no mobility seen in teeth. There is no calcifying focal spot in panoramic radiographs while there is in CT. It maintains the speed of development. Calcified focal spot demonstrates slow development.[20]


  Conclusion Top


CEOT is locally invasive odontogenic tumor with a high recurrence rate. Lesions of CEOT in the maxilla should be treated as soon as possible because these tumors usually develop and invade to the vital structures quicker than mandibular tumors. Surgical options have varieties such as enucleation, curettage, and radical resection. However, large extent recurrence of the tumor occurs due to insufficient surgical procedures. After surgery, patients should need periodic clinical and radiographic follow-ups.

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.



 
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Tarsitano A, Agosti R, Marchetti C. The diagnostic and surgical management of a multifocal calcifiyng epithelial odontogenic tumor in the mandible and maxilla associated with a squamous odontogenic tumor:First reported case in the literature. Oral Surg Oral Med Oral Pathol Oral Radiol 2012;113:e6-11.  Back to cited text no. 12
    
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Mujib BR, Kulkarni PG, Lingappa A, Jahagirdar A, Soman C. An atypical presentation of pindborg tumor in anterior maxilla. Dent Res J (Isfahan) 2012;9:495-8.  Back to cited text no. 13
    
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Anavi Y, Kaplan I, Citir M, Calderon S. Clear-cell variant of calcifying epithelial odontogenic tumor: Clinical and radiographic characteristics. Oral Surg Oral Med Oral Pathol Oral Radiol Endod 2003;95:332-9.  Back to cited text no. 14
    
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Gopalakrishnan R, Simonton S, Rohrer MD, Koutlas IG. Cystic variant of calcifying epithelial odontogenic tumor. Oral Surg Oral Med Oral Pathol Oral Radiol Endod 2006;102:773-7.  Back to cited text no. 16
    
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    Figures

  [Figure 1], [Figure 2], [Figure 3], [Figure 4], [Figure 5], [Figure 6], [Figure 7], [Figure 8], [Figure 9], [Figure 10], [Figure 11], [Figure 12], [Figure 13], [Figure 14], [Figure 15], [Figure 16]



 

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