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Year : 2018  |  Volume : 61  |  Issue : 2  |  Page : 290-291
Peripheral variant of calcifying epithelial odontogenic tumor with clear cell changes: An enigma


Department of Oral and Maxillofacial Pathology, NIMS Dental College, Jaipur, Rajasthan, India

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Date of Web Publication20-Apr-2018
 

How to cite this article:
Bajpai M. Peripheral variant of calcifying epithelial odontogenic tumor with clear cell changes: An enigma. Indian J Pathol Microbiol 2018;61:290-1

How to cite this URL:
Bajpai M. Peripheral variant of calcifying epithelial odontogenic tumor with clear cell changes: An enigma. Indian J Pathol Microbiol [serial online] 2018 [cited 2020 Feb 21];61:290-1. Available from: http://www.ijpmonline.org/text.asp?2018/61/2/290/230564




Calcifying epithelial odontogenic tumors (CEOTs) were first described by Pindborg in 1955.[1] CEOTs are rare odontogenic neoplasms of the jaws that account for <1% of all odontogenic tumors. The peripheral (extraosseous) counterpart of CEOT is an extremely rare lesion accounts only for 6% of all CEOTs.[2] Very few cases of peripheral CEOT have been reported in the literature. Moreover, <5 cases of peripheral CEOT with clear cell changes have been found in an exhaustive literature review.

An otherwise healthy 34-year-old male presented to the department of oral medicine and radiology for the evaluation of soft-tissue growth in her upper front region of the jaw for 9 months. The family history and past medical history of the patient was nonrelevant to the presenting symptom. Intraoral examination revealed a pink-colored swelling with relation to the lingual surface of tooth #11 extending anteroposteriorly #12 measuring about 2 cm × 3 cm. The swelling was soft to firm on palpation. No signs of ulceration and discharge were found. Intraoral periapical radiograph revealed a localized loss of lamina dura with relation to #11 and #12. Based on the clinical and radiological features, a provisional diagnosis peripheral ossifying fibroma was given. The lesion was excised under local anesthesia and the resected tissue was sent to the department of oral and maxillofacial pathology for expert comments. The follow-up period of 6 months was uneventful and showed a satisfactory healing.

Histopathological examination of the soft-tissue section showed a well-encapsulated lesion showed hyperplastic epithelium with hyalinized connective tissue stroma [Figure 1]a comprised numerous islands of polyhedral squamous epithelial cells with hyperchromatic nucleus, prominent intercellular bridges. Few islands of polyhedral cells reveal clear cell changes [Figure 1]b. Areas of extracellular amyloid deposition were seen [Figure 1]c with abundant calcified masses showing characteristic Liesegang rings [Figure 1]d.
Figure 1: (a) A well-encapsulated lesion with hyperplastic epithelium and hyalinized connective tissue stroma showing numerous eosinophilic calcified masses (H and E, ×20). (b) Numerous islands of polyhedral squamous cells with hyperchromatic nuclei and prominent intercellular bridges. Few islands of polyhedral cells revealing clear cell changes (H and E, ×40). (c) Extensive deposition of extracellular, eosinophilic amyloid-like material (H and E, ×20). (d) Extensive calcifications in the form of characteristic Liesegang ring pattern (H and E, ×40)

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The tissue was also stained with Congo red that revealed abundant amyloid type material and focal areas of calcification in the form of Liesegang rings [Figure 2]a. Apple-green birefringence was observed under polarized microscopy [Figure 2]b.
Figure 2: (a) Picture shows amyloid positivity with Congo red stain. (b) Polarized microscopy reveals apple-green birefringence for amyloids

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Based on histopathology, a final diagnosis of peripheral CEOT with clear cell changes was rendered.

Peripheral odontogenic tumors are unusual; very few cases of peripheral CEOT have been reported in the literature to date.[3] Peripheral CEOTs are histopathologically characterized by a well-unencapsulated lesion with islands of polyhedral cells with hyperchromatic nuclei and prominent intercellular bridges, the epithelium may or may not show dysplastic features, accompanied by abundant amyloid-like structures with calcification in the form of Liesegang rings.[1] Peripheral CEOT with clear cell changes is an extremely rare lesion. The present case reveals clear cell changes in the islands of polyhedral squamous cells. Afrogheh et al.,[2] in 2014, recommended two variants of CEOT, namely, CEOT with clear cell changes and CEOT with clear Langerhans cells. The present case also elucidated the importance of Congo red staining for amyloid demonstration. The differential diagnoses of peripheral CEOT with clear cell changes include clear cell odontogenic carcinoma (CCOC) and clear-cell carcinoma (CCC).[4] The differentiation is utmost important since CCC and CCOC are malignant tumors and requires aggressive treatment approach. We further suggest that peripheral CEOTs should be included in the differential diagnosis of asymptomatic gingival growths.

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Conflicts of interest

There are no conflicts of interest.



 
   References Top

1.
Pindborg JJ. Calcifying epithelial odontogenic tumors. Acta Pathol Microbiol Scand 1955;111:71.  Back to cited text no. 1
    
2.
Afrogheh A, Schneider J, Mohamed N, Hille J. Calcifying epithelial odontogenic tumour with clear langerhans cells: A novel variant, report of a case and review of the literature. Head Neck Pathol 2014;8:214-9.  Back to cited text no. 2
    
3.
Bajpai M, Pardhe N. Peripheral ameloblastoma with mixed histological patterns. Cukurova Med J 2015;40:151-5.  Back to cited text no. 3
    
4.
Kauzman A, Tabet JC, Stiharu TI. Hyalinizing clear cell carcinoma: A case report and review of the literature. Oral Surg Oral Med Oral Pathol Oral Radiol Endod 2011;112:e26-34.  Back to cited text no. 4
    

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Correspondence Address:
Manas Bajpai
Department of Oral and Maxillofacial Pathology, NIMS Dental College, Jaipur, Rajasthan
India
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Source of Support: None, Conflict of Interest: None


DOI: 10.4103/IJPM.IJPM_679_17

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    Figures

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