|Year : 2009 | Volume
| Issue : 2 | Page : 79-82
Calcifying epithelial odontogenic cyst with odontome in left mandible
R Kamala, Abhishek Sinha, Mamta Sharma
Department of Oral Medicine and Radiology, Sardar Patel Postgraduate Institute of Dental and Medical Sciences (SPPGIDMS), Lucknow, India
|Date of Web Publication||1-Dec-2009|
Department of Oral Medicine and Radiology, Sardar Patel Postgraduate Institute of Dental and Medical Sciences (SPPGIDMS), Lucknow
Source of Support: None, Conflict of Interest: None
| Abstract|| |
Calcifying epithelial odontogenic cyst is a mixed odontogenic benign tumor but most of the cases present cystic characteristics. We present a case report of calcifying epithelial odontogenic cyst with odontoma in a 20-year-old male who presented with a painless unilateral swelling of the jaw. The lesion involved an impacted 37 which was displaced to the lower border of the mandible and a calcified mass that was within the cystic lesion that was recognized as odontoma. The lesion was surgically removed along with 36 and 37 was allowed to erupt.
Keywords: Calcifying epithelial odontogenic cyst, ghost cells, odontocalcifying odontogenic cyst, odontoma
|How to cite this article:|
Kamala R, Sinha A, Sharma M. Calcifying epithelial odontogenic cyst with odontome in left mandible. J Indian Acad Oral Med Radiol 2009;21:79-82
|How to cite this URL:|
Kamala R, Sinha A, Sharma M. Calcifying epithelial odontogenic cyst with odontome in left mandible. J Indian Acad Oral Med Radiol [serial online] 2009 [cited 2019 Apr 20];21:79-82. Available from: http://www.jiaomr.in/text.asp?2009/21/2/79/57893
| Introduction|| |
The cysts and the tumors that originate from the odontogenic tissues represent a diverse group of lesions that reflect the deviations from the normal pattern of odontogenesis. The calcifying epithelial odontogenic cyst derived from the odontogenic epithelial remnants within either the jaws or the gingival.  This distinct entity was first described by Gorlin et al. in 1962. , The condition was previously described in the German literature in 1932 by Rywkind. 
In 1992 the World Health Organization classified COC within the groups of neoplasms and tumors that originate from the odontogenic tissues but confirmed that most of the cases are non-neoplastic. 
The lesion is a mixed odontogenic benign tumor and although most of the cases present cystic characteristics, a few are of the solid type and its malignant transformation is well documented.  The COCs may occur in association with other odontogenic tumors, the most common is the odontoma, occurring in about 24% of the cases.  The COC appears as a painless, slow-growing tumor affecting equally the maxilla and mandible, with predilection for the anterior segment (incisor/canine area). It generally affects young adults in the third to fourth decade without gender predilection.
Radiographically, COC is usually a mixed lesion, appears as a unilocular or multilocular well-defined radiolucency with irregular calcified bodies of varying sizes, and it may be associated with an odontoma or an unerupted tooth. 
The most notable histopathological feature is the presence of the ghost epithelial cells with the propensity to calcify. 
| Case Report|| |
The present report describes a case of calcifying epithelial odontogenic cyst with odontoma in a 20-year-old male who reported to the Oral Medicine and Radiology Department of Sardar Patel Postgraduate Institute of Dental and Medical Sciences, with a chief complaint of swelling on the left side of the face of 15 months duration. Patient revealed in history that swelling started gradually 15 months back and increased to attain the present size. It was not associated with pain, discharge and numbness. No associated signs and symptoms were present. His past medical and dental history were non-contributory. General examination revealed a moderately built and nourished individual of normal gait with vital signs within the normal range.
Extraoral inspection revealed an oval-shaped swelling measuring 5 cm x 6 cm in diameter present in the left body and ramus of the mandible extending superiorly up to the left zygomatic arch, inferiorly 1 cm below the inferior border of the mandible, medially 2 cm away from left angle of mouth, laterally posterior to the posterior border of ramus [Figure 1]. Skin over the swelling was slightly stretched with no secondary changes. On palpation it was non-tender, hard in consistency, no localized rise in temperature was noticed, non-fluctuant, and skin over the swelling was easily pinchable and appeared normal. Swelling was fixed to the underlying body structures.
Intraoral examination revealed a diffused swelling present in the lower left posterior jaw region measuring 23 cm, with a normal-appearing mucosa extending anteriorly up to the gingivobuccal sulcus of 35, posteriorly retromolar trigone laterally causing obliteration of buccal vestibule [Figure 2]. Mucosa overlying the lesion was intact and teeth in the vicinity showed no mobility, discoloration, tenderness and responded positive to the vitality tests, 36 was slightly lingually tilted and 37 was missing. Aspiration yielded a brownish-yellow-colored fluid mixed with blood [Figure 3]. Various differential diagnoses were included such as dentigerous cyst, mural ameloblastoma, odontogenic keratocyst, calcifying epithelial odontogenic cyst.
Radiographic investigations: mandibular cross-sectional occlusal radiograph revealed bicortical plate expansion and thinning of the cortex and lingually tilted 36 [Figure 4]. Panoramic radiograph revealed a unilocular radioluceny measuring 4 x 5cm in diameter with a sclerotic border extending from the mesial of 36 involving the left body and ascending ramus involving the left coronoid and condyle with a radio-opaque mass seen in the anterior aspect of ramus and impacted 37, the crown of which was surrounded by another radioluceny and the tooth was displaced to the inferior border of the mandible [Figure 5].
The surgical management included enucleation of the cystic lesion with removal of the odontomes and extraction of 36 under local anesthesia [Figure 6]; 37 was preserved and allowed to erupt.
Photomicrograph revealed a lining of epithelium resembling odontogenic apparatus and numerous ghost cells. Basal cells resembled ameloblasts, suprabasal cells resembled stellate reticulum [Figure 7]. Ground section of calcified masses showed irregularly arranged enamel, dentine, and pulp spaces.
In the follow-up done after six months panoramic view revealed slight migration of the 37 [Figure 8]a and in one-year follow-up OPG revealed normal erupting of 37 [Figure 8]b.
History, clinical examination, aspiration, radiographic and histopathological evaluation confirmed the diagnosis of calcifying epithelial odontogenic cyst with complex odontoma.
| Discussion|| |
This case report is in agreement with the literature finding that COC occurs predominantly as an intraosseous lesion.  Hirshberg et al. proposed the term "odontocalcifying odontogenic cyst" for this variant.  They observed that this variant is more prevalent in females, with a mean age of 16 years at discovery and most cases were located in the maxilla. , The findings of our case are not in accordance with the gender and site predilection. Radiographically, it appears as a mixed radiolucent radio-opaque lesion with a well-circumscribed border which is encountered in the majority COCs with odontomas. ,
The defining microscopic feature of this lesion is the presence of variable numbers of altered epithelial cells without nuclei. They tend to be lightly eosinophilic and retain the basic cell outline, and have been termed "ghost cells".  Several possibilities suggest that the COC develops secondarily from the odontogenic epithelium that participates in the formation of the odontoma. However, it also has been suggested that the odontoma develops secondarily from the lining epithelium of the COC.
The COC with odontoma should be treated conservatively by surgical enucleation because recurrences are very uncommon. 
| Conclusion|| |
COC can be found in association with odontogenic tumors like odontoma, ameloblastomas, ameloblastic fibro-odontoma, odontoameloblastic tumor, calcifying epithelial odontogenic tumor and adenomatoid odontogenic tumor. In the present case the lesion was associated with an odontome.
The association of unerupted teeth is a common radiographic feature of COC. The frequency of enveloped teeth is reported to approximate 32%, and a lesion involving an impacted tooth may simulate a dentigerous cyst when viewed on radiographs. In the present case the lesion was treated conservatively by surgical enucleation and extraction of 36, and 37 was preserved and allowed to erupt normally with normal bone healing and no recurrence noticed after one year and further follow-up was advised.
| Acknowledgment|| |
I would like to convey my special thanks to Dr. T.S. Bastian, Prof& Head, Department of Oral and Maxillofacial Pathology, Sardar Patel Postgraduate Institute of Dental and Medical Sciences, for his valuable help in slide preparation.
| References|| |
|1.||Gallana-Alvarez S, Mayorga-Jimenez F, Torres-Gσmez FJ, Avellα-Vecino FJ, Salazar-Fernandez C. Calcifying odontogenic cyst associated with complex odontoma: case report and review of the literature. Med Oral Patol Oral Cir bucal 2005;10:243-7. |
|2.||Iida S, Fukuda Y, Ueda T, Aikawa T, Arizpe JE, Okura M. Calcifying odontogenic cyst: radiologic findings in 11 cases. Oral Surg Oral Med Oral Pathol Oral Radiol Endod 2006;101:356-62. |
|3.||Erasmus JH, Thompson IO, van Rensburg LJ, van der Westhuijzen AJ. Central calcifying odontogenic cyst. A review of the literature and the role of advanced imaging techniques. Dentomaxillofac Radiol 1998;27:30-35. |
|4.||Johnson A 3rd, Fletcher M, Gold L, Chen SY. Calcifying odontogenic cyst: a clnicopathologic study of 57 cases with immunohistochemical evaluation for cytokeratin. J Oral Maxillofac Surg 1997;55:679-83. |
|5.||Moleri AB, Moreira LC, Carvalho JJ. Comparative morphology of 7 new cases of calcifying odontogenic cysts. J Oral Maxillofac Surg 2002;60:689-96. |
|6.||Pistσia GD, Gerlach RF, dos Santos JC, Montebelo Filho A. Odontoma producing intraosseous calcifying odontogenmic cyst: case report. Braz Dent J 2001;12:67-70. |
|7.||Rushton VE, Horner K. Calcifying odontogenic cyst- a characteristic CT finding. Br J Oral Maxillofac Surg 1997;35:196-8. |
|8.||Souza LN, Souza AC, Gomes CC, Loyola AM, Durighetto AF Jr, Gomez RS, et al. Conservative treatment of calcifying odontogenic cyst: report of 3 cases. J Oral Maxillofac Surg 2007;65:2353-6. |
[Figure 1], [Figure 2], [Figure 3], [Figure 4], [Figure 5], [Figure 6], [Figure 7], [Figure 8]