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CASE REPORT
Year : 2014  |  Volume : 32  |  Issue : 2  |  Page : 149-151
 

Calcifying epithelial odontogenic tumor, a rare presentation in children: Two case reports


1 Department of Pedodontics, Institute of Dental Sciences, Siksha O Anusandhan University, Ghatikia, Bhubaneswar, Odisha, India
2 Department of Oral Pathology and Microbiology, Institute of Dental Sciences, Siksha O Anusandhan University, Ghatikia, Bhubaneswar, Odisha, India
3 Department of Oral Medicine and Radiology, Institute of Dental Sciences, Siksha O Anusandhan University, Ghatikia, Bhubaneswar, Odisha, India
4 Department of Oral Medicine and Radiology, Krishnadevaraya College of Dental Sciences and Hospital, Bengaluru, Karnataka, India

Date of Web Publication17-Apr-2014

Correspondence Address:
Samapika Routray
Department of Oral Pathology and Microbiology, Institute of Dental Sciences, Siksha O Anusandhan University, K-8, Ghatikia, Bhubaneswar -751 003, Odisha
India
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Source of Support: None, Conflict of Interest: None


DOI: 10.4103/0970-4388.130977

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   Abstract 

Calcifying epithelial odontogenic tumor (CEOT) is a rare and benign odontogenic neoplasm that affects the jaws. It is certainly an atypical instance to find this tumor in children. Here, we present two case reports of CEOT presenting in mandible of a 12- and 13-year-old female child, respectively. CEOT have been reported to show features of malignant transformation also.


Keywords: CEOT, Children, Female, Mandible, Unusual


How to cite this article:
Mohanty S, Mohanty N, Routray S, Misra SR, Vasudevan V. Calcifying epithelial odontogenic tumor, a rare presentation in children: Two case reports. J Indian Soc Pedod Prev Dent 2014;32:149-51

How to cite this URL:
Mohanty S, Mohanty N, Routray S, Misra SR, Vasudevan V. Calcifying epithelial odontogenic tumor, a rare presentation in children: Two case reports. J Indian Soc Pedod Prev Dent [serial online] 2014 [cited 2020 Feb 21];32:149-51. Available from: http://www.jisppd.com/text.asp?2014/32/2/149/130977



   Introduction Top


The calcifying epithelial odontogenic tumor (CEOT) was first introduced by Dr J J Pindborg in 1955, referred to as the Pindborg tumor. [1] It is a rare entity and represents less than 1% of all odontogenic tumors possibly of stratum intermedium origin. This idea is based on the morphological similarity of the tumor cells to the normal cells of the stratum intermedium, and high activity of alkaline phosphatase and adenosine triphosphate. A different hypothesis in which this tumor may arise from remnants of the primitive dental lamina found in the initial stage of odontogenesis also exists. It occurs mostly in second to sixth decade of life (40 years as mean) and bears no gender predilection. [2] Clinically, CEOT manifests as an intraosseous lesion (central type) in the majority of cases (95%). Extraosseous or peripheral lesions account for less than 5% of cases. [3] The purpose of this article is to report two cases of CEOT in pediatric patients that is rarely observed and discuss the clinical behavior, radiologic appearance, and histologic features for better surgical management. Patient was then referred to Department of Oral Surgery for treatment and management. Later hemimanidibulectomy was done with reconstruction.


   Case Report 1 Top


A female aged 12 years visited the Oral Diagnosis Discipline complaining of pain on chewing food. The patient presented a history of broken tooth and swelling that gradually increased in size since 2 years in relation to the right lower jaw back region. On clinical inspection, a diffuse bony hard swelling over the right body of mandible revealed with palpable and tender regional lymph nodes [Figure 1]a, b. Intraorally a bony hard non-tender swelling in relation to 43 till 46 region with buccolingual expansion of cortical plates retained 84, 85, and displaced 43 was observed [Figure 1]c. Orthopantomogram revealed a diffuse radio-opaque area interspersed with radiolucencies; the radio-opaque mass is of varying radiodensities alongwith impacted 44 next to the lower border of mandible [Figure 2]. Incisional biopsy was done under local anesthesia, and the section revealed presence of sheets of polyhedral epithelial cells in a fibrous connective tissue stroma with prominent intercellular bridges and multiple areas of calcifications [Figure 3]. The overall features were suggestive of CEOT.
Figure 1: a, b, c Clinical and intraoral photomicrographs of Case 1

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Figure 2: OPG shows diffuse radio-opaque area interspersed with radio-lucencies the radio-opaque mass is of varying radiodensities alongwith impacted 44 next to the lower border of mandible

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Figure 3: Sheets of polyhedral epithelial cells in a fi brous connective tissue stroma with prominent intercellular bridges and multiple areas of calcifi cations

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   Case Report 2 Top


A female aged 13 years visited the Oral Diagnosis Discipline presenting a swelling in the lower jaw with discomfort and difficulty during taking food. Clinical examination revealed a relatively large diffuse, bony hard, and non-tender swelling involving the body and ramus of mandible with thinning of lower border [Figure 4]a, b. Intraoral inspection showed a well-defined, non-tender, firm swelling involving 34 till 37 region with expansion and distortion of the buccal cortical plates and minimum involvement lingually [Figure 4]c. The patient reported that the lesion developed in past 1 year. Radiographic examination revealed a large radiolucent lesion extending from 33 till 37 region with multiple irregular radio-opaque foci [Figure 5]a, b, c]. Resorption of roots of second premolar and first molar with extensive thinning of lower border of left side of mandible was observed. An incisional biopsy was undertaken and specimen was sent to oral pathology laboratory. Histopathology showed presence of sheets of polyhedral epithelial cells in a fibrous connective tissue stroma, showing cellular pleomorphism. There were multiple areas of globular calcifications. At places homogenous eosinophilic areas and circumscribed area of clear cells are present. The features were confirmative of CEOT. Patient was then referred to Department of Oral Surgery for treatment and management. The patient underwent naso-endothelial intubation with flexometalic number 6 tube. Risdon incision was marked taking into consideration of the course marginal mandibular nerve. Complete lesion was exposed and resection was done from midline to the left condyle. Reconstruction was done with 2.7-mm titanium reconstruction plate and later kept for delayed vascularized fibula flap [Figure 6]. Wound was closed into layers. Postoperative check up was done after 7 days.
Figure 4: a, b, c Clinical and intraoral photomicrographs of Case 2

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Figure 5: a, PNS, b, oblique lateral, and c, OPG: A large radiolucent lesion extending from 33 to 37 region with multiple irregular radio-opaque foci and resorption of roots of second premolar and fi rst molar with extensive thinning of lower border of left side of mandible

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Figure 6: Reconstruction was done with 2.7-mm titanium reconstruction plate

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   Discussion Top


The uniqueness of two cases in this report is based on the age of occurrence of the tumor. The confirmatory diagnosis of CEOT is based on the radiographic and histopathological correlation after the clinical diagnosis was made. It is usually associated with an impacted tooth and in initial stages appears radiolucent mimicking a dentigerous cyst. In aggressive lesions, osseous destruction and the tumoral calcification is observed, giving it a honeycomb appearance. [3] Histopathological examination revealing polyhedral neoplastic cells, which have abundant eosinophilic finely granular cytoplasm with nuclear pleomorphism and prominent nucleoli. Most of the cells are arranged in anastomosing sheet like masses. An extracellular eosinophilic homogenous material staining like amyloid is characteristic of this tumor with concentric calcific deposits called Liesgang ring. [4],[5],[6] A recent study confirmed PTCH1 mutations in the CEOT indicating at involvement of mutations of the various proteins in sonic hedgehog signaling pathway, which regulates embryonic development and participates in odontogenesis through mediation of epithelial-mesenchymal interactions. [7]

The treatment plan in cases of CEOT depends on multiple factors such as size and location of neoplasm, general condition of patient, and operator skill. If the tumor is small intrabony mandibular lesions with well-defined borders then simple enucleation or curettage followed by judicious removal of a thin layer of bone adjacent to the tumor is enough. Large tumors have to be approached by segmental resection and reconstruction using grafting or distraction osteogenesis. As these tumors are not encapsulated, it is commonly agreed that the resection should include a safe margin of clinically and radiographically healthy bone. [8] Malignant transformation and metastasis is rare though reported in literature.

In our cases, after the preanesthetic checkup, the medication was given. Patient reported here underwent hemimandibulectomy in both the cases and there has been no recurrence on 6 month follow-up. The Case 2 patient underwent hemimandibulectomy and reconstruction was done with titanium reconstruction plate. Though recurrence rates for CEOT vary from 10 to 15%, requiring periodic clinical and radiographic follow-up, our cases did not show after a long-term follow-up also. A relatively higher recurrence rate of 22% has been reported for the clear cell variant. [9] The relevance of reporting both the cases is that considering the age factor, CEOT was seen in these two young female child patients, and it is mandatory for the surgeon to have an aesthetic approach for future prospective.

 
   References Top

1.Pindborg JJ. Calcifying epithelial odontogenic tumour. Acta Pathol Microbiol Scand 1955;111:71.  Back to cited text no. 1
    
2.Ai-Ru L, Zhen L, Jian S. Calcifying epithelial odontogenic tumors: A clinicopathologic study of nine cases. J Oral Pathol 1982;11:399-406.  Back to cited text no. 2
[PUBMED]    
3.Goode RK. Calcifying epithelial odontogenic tumor. Oral Maxillofac Surg Clin North Am 2004;16:323-31.  Back to cited text no. 3
[PUBMED]    
4.Smith RA, Roman RS, Hansen LS, Lundell WJ, Riley RW. Oral surgery program university of california, san francisco. J Oral Surg 1977;35:160-6.  Back to cited text no. 4
[PUBMED]    
5.Marx RE, Stern D. Oral and maxillofacial pathology: A rationale for diagnosis and treatment. Chicago: Quintessence; 2003.  Back to cited text no. 5
    
6.Patino B, Fernandez-Alba J, Garcia-Rozado A, Martin R, Lopez-Cedrun JL, Sanroman B. Calcifying epithelial odontogenic (pindborg) tumor: A series of 4 distinctive cases and a review of the literature. J Oral Maxillofac Surg 2005;63:1361-8.  Back to cited text no. 6
    
7.Peacock ZS, Cox D, Schmidt BL. Involvement of PTCH1 mutations in the calcifying epithelial odontogenic tumor. Oral Oncol 2010;46:387-92.  Back to cited text no. 7
    
8.Tarsitano A, Agosti R, Marchetti C. The diagnostic and surgical management of a multifocal calcifying 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. 8
    
9.Hicks MJ, Flaitz CM, Wong ME, McDaniel RK, Cagle PT. Clear cell variant of calcifying epithelial odontogenic tumor: Caser report and review of the literature. Head Neck 1994;16:272-7.  Back to cited text no. 9
    


    Figures

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



 

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    Abstract
   Introduction
   Case Report 1
   Case Report 2
   Discussion
    References
    Article Figures

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