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 Table of Contents  
CASE REPORT
Year : 2019  |  Volume : 31  |  Issue : 2  |  Page : 181-183

Calcifying cystic odontogenic tumor of anterior mandible - A case report with series of follow up radiographs


1 Senior Lecturer, Department of Oral Medicine and Radiology, Annoor Dental College and Hospital, Muvattupuzha, Ernakulam, Kerala, India
2 Prof and HOD, Department of Oral Medicine and Radiology, Vydehi Institute of Dental Sciences and Research Sciences, White Field, Bengaluru, Karnataka, India
3 Reader, Department of Oral Medicine and Radiology, Vydehi Institute of Dental Sciences and Research Sciences, White Field, Bengaluru, Karnataka, India
4 Consultant Maxillofacial Radiologist, Pune, India

Date of Submission30-Jan-2019
Date of Acceptance04-Mar-2019
Date of Web Publication24-Jun-2019

Correspondence Address:
Dr. Hima John
Department of Oral Medicine and Radiology, Annoor Dental College and Hospital, Muvattupuzha, Ernakulam - 686 673, Kerala
India
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Source of Support: None, Conflict of Interest: None


DOI: 10.4103/jiaomr.jiaomr_25_19

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   Abstract 


The calcifying odontogenic cyst was first described by Gorlin et al. who were impressed by the presence of numerous ghost cells. Currently, this particular entity is known as calcifying cystic odontogenic tumor (CCOT). Since its recognition, the lesion is seen occurring both peripherally and centrally in the jaws. Its origin, pathogenesis, and histopathological variations have evoked considerable discussions in the past which has resulted in formulation of various classifications. This paper highlights a case of CCOT in the anterior mandible, the characteristic features, treatment and follow-up are discussed, emphasizing on the series of follow-up radiographs.

Keywords: Calcifying cystic odontogenic tumor, calcifying epithelial odontogenic cyst, ghost cells


How to cite this article:
John H, Padmashree S, Shilpa P S, Innus SZ. Calcifying cystic odontogenic tumor of anterior mandible - A case report with series of follow up radiographs. J Indian Acad Oral Med Radiol 2019;31:181-3

How to cite this URL:
John H, Padmashree S, Shilpa P S, Innus SZ. Calcifying cystic odontogenic tumor of anterior mandible - A case report with series of follow up radiographs. J Indian Acad Oral Med Radiol [serial online] 2019 [cited 2019 Jul 21];31:181-3. Available from: http://www.jiaomr.in/text.asp?2019/31/2/181/261087




   Introduction Top


The calcifying cystic odontogenic tumor (CCOT), which represents 2% of all odontogenic tumors was first described as a distinctive cystic entity by Gorlin et al., in 1962, was thought to be a possible oral analogue of the dermal calcifying epithelioma of Malherbe.[1] Over the years, various researchers have found that the calcifying odontogenic cyst (COC) has various entities ranging from simple cysts to neoplasms. It was classified as a benign odontogenic tumor, with the SNOMED code 9301/0, in the World Health Organization's (WHO) publication, Histological Typing of Odontogenic Tumours by Kramer et al.[2]

There has been a complete re-evaluation of this lesion by many authors with many classifications being put forth. One such major conclusion was put forth by Praetorius et al. was that it comprised two entities: a cyst and a neoplasm.[3] Several classifications have been proposed depending upon the variety of histopathologic features as well as biologic behaviors. Some COCs appear to represent simple cysts whereas others have no cystic features and are regarded as neoplastic.[4]


   Case Report Top


A 32-year-old male patient presented to the department of oral medicine and radiology with the chief complaint of dull aching type of pain and swelling in the lower front tooth region since 3 years. Patient gave history of trauma to the chin region from a hand pump 5 years ago after which he had mild pain and swelling in the chin region which regressed after about a week.

On extra oral examination, on inspection and palpation no abnormality was detected. On intraoral inspection, diffuse swelling was noted in the labial vestibule irt 31,32,41,42,43 with obliteration of the labial vestibule. Surface appeared smooth and pink, with an area of blanching noted irt 31,41. Hard tissue examination -41 appeared discolored. The swelling was bony hard in consistency except in the region of 41 were it was soft and fluctuant [Figure 1]a. 41 was tender on vertical percussion. Based on the above features, a clinical diagnosis of Radicular cyst irt 41 was given. Electric Pulp Vitality test was done, no response irt 41,33. Delayed response irt 31,32,42,43 was noted. FNAC done–the aspirate was dark brown in color.
Figure 1: (a) Intraoral photograph showing swelling in the labial vestibule. (b) Mandibular occlusal radiograph showing the buccal and lingual cortical plate expansion. (c) OPG revealed a well-defined radiolucency with scalloped border and sclerotic margin in the mandibular anterior region

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Intra oral periapical radiographs taken irt 31,32,33,41,42 revealed well-defined radiolucency involving the middle and apical third of the root, surrounded by a sclerotic border. Occlussal radiograph revealed the presence of well-defined radiolucency noted extending from the mesial aspect of 37, crossing the midline till the mesial aspect of 46. Expansion of the buccal and lingual cortical plate was appreciated [Figure 1]b. OPG revealed a well-defined radiolucency with scalloped border and sclerotic margin in the mandibular anterior region, extending from the mesial aspect of 37, crossing the midline till the mesial aspect of 46 [Figure 1]c. Computed tomography and 3D reconstruction revealed the presence of expansion and perforation of the buccal cortical plate irt 31,32,34,41,42,44,45 with the HU ranging from + 40 to + 120 in the lesional tissue. [Figure 2]a and [Figure 2]b. Based on the above features, a radiographic differential diagnosis of traumatic bone cyst and central giant cell granuloma were given. Incisional biopsy was nonspecific; therefore, surgical enucleation followed by curettage was done and the specimen was sent for histopathological evaluation. Root canal treatment was done for 31,32,33,34,35,41,42,43,44,45.
Figure 2: (a) CT Axial section depicting the expansion and extent of the lesion. (b) The 3D reconstruction of CT images. (c) The histopathological section showing the presence of ghost cells (white arrow)

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Histopathological evaluation revealed the presence of features typical for calcifying odontogenic tumor with the presence of the keratinized ghost cells [Figure 2]c. Based on these features, the lesion was diagnosed as intraosseous cystic variant of calcifying cystic odontogenic tumor (CCOT). [Figure 3]a and [Figure 3]b show the 1 month postoperative OPG and occlussal radiograph and [Figure 3]c shows the 6 months postoperative occlussal radiograph.
Figure 3: (a and b) 1 month postop radiographs. (c) 6 months postop mandibular occlusal radiograph showing ample bone deposition

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   Discussion Top


CCOT was first described by Rywkind in 1932 who reported a lesion of the jaw which resembled cholesteatoma of the ear. He henceforth called it as cholesteatoma of the jaw.[5],[6] In 1946, it was described as a strange variant of ameloblastoma by Thoma and Goldman. Tomich reported incidence of CCOT as less than two cases per year and recorded a total of 51 cases in 34 years. In 1981 Preatorius et al. proposed that calcifying odontogenic cyst comprises of two entities: a cyst and a neoplasm.[7],[8] In 2008, an international collaborative study distinguished four variants of CCOT. They employed a more objective, comprehensive, and useful classification that would include all the so called COC subtypes (both cystic and solid) under the 2005 WHO guidelines. It was as follows: CCOT type 1 (simple cystic), CCOT type 2 (odontomas-associated), CCOT type 3 (ameloblastomatous proliferation), and CCOT type 4 (associated with benign odontogenic tumors other than odontoma).[9]

CCOT is common in the second decade of life. Preatorius and Buchner have drawn attention to a bimodal age distribution with a second peak in the 6--7th decade of life.[4],[10] There are no particular predilections for either the maxilla or mandible. Canine--incisor portion is the site of occurrence in both jaws and the same was seen in our case also.[10]

Studies point out that central COCs occur more common than peripheral lesions by a 3:1 ratio, and they are usually diagnosed in the second decade of life, while the peripheral ones are usually noted after 50 years. In our case, however, it was diagnosed in the 4th decade of life.[11]

Radiographically, CCOT is usually a mixed radiopaque--radiolucent lesion. It may be unilocular or multilocular. It has been noted in association with impacted teeth in about 10--32% of the cases. Displacement of teeth and resorption of the roots of the adjacent teeth are frequent findings.[12],[13] The same features were noted in this case too. It is thought to occur from the proliferation of remnants of dental lamina. Many authors believe that ghost cells represent normal or abnormal keratinization.

The treatment of cystic lesion involves enucleation with long-term follow up. Recurrence is noted if incomplete cyst removal is done. Prognosis is good for cystic CCOT and less certain for neoplastic variants. This case is currently under follow-up and has remained symptom free for the past one year.


   Conclusion Top


Our case here represents the typical cystic variant of the CCOT which had fully occupied the anterior mandible. This case shows how the appropriate diagnosis and management of a case can prove beneficial for the patient which can be appreciated in the postoperative series of radiographs over a year's time.

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.
Gorlin RJ, Pindborg JJ, Clausen FP, Williamson JJ, Hansen LS. The calcifying odontogenic cyst. A possible analogue of the cutaneous calcifying epithelioma of Malherbe. Oral Surg Oral Med Oral Pathol 1962;15:1235-43.  Back to cited text no. 1
    
2.
Kramer IRH, Pindborg JJ, Shear M. Histological Typing of Odontogenic Tumours. Berlin: Springer Verlag; 1992.  Back to cited text no. 2
    
3.
Prætorius F, Ledesma-Montes C. Calcifying cystic odontogenic tumour. In: Barnes L, Eveson JW, Reichart P, Sidransky D, editors. Pathology and Genetics. Head and Neck Tumours. Lyon: World Health Organization Classification of Tumours, IARC Press; 2005. p. 313.  Back to cited text no. 3
    
4.
Shear M, Speight P. Cysts of the Oral and Maxillofacial Regions. 4th ed. Blackwell-Munksgaard; 2007. p. 100-7.  Back to cited text no. 4
    
5.
Gupta N, Gupta S. Ameloblastomatous calcifying odontogenic cyst: A rare clinicopathologic entity. Int J Head Neck Surg 2011;2:115-8.  Back to cited text no. 5
    
6.
Santos T, Frota R, Andrade E, Oliveira E. Calcifying cystic odontogenic tumor radiographically mimicking a sinus mucocele. J Craniofac Surg 2013;24:134-6.  Back to cited text no. 6
    
7.
Hanttash A, Oncul A, Aktaş U. Calcifying cystic odontogenic tumor presenting as a dentigerous cyst: A case report with literature review. Int J Oral Maxillofac Pathol 2016;6:11-8.  Back to cited text no. 7
    
8.
Estrela C, Decurcio DA, Silva JA, Mendonça EF, Estrela CRA. Persistent apical periodontitis associated with a calcifying odontogenic cyst. Int Endod J 2009;42:539-45.  Back to cited text no. 8
    
9.
Zornosa X, Muller S. Calcifying cystic odontogenic tumor. Head Neck Pathol 2010;4:292-4.  Back to cited text no. 9
    
10.
Menat S, Shylaja M, Attur K, Goyal K. Ameloblastomatous CCOT: A case report of a rare variant of CCOT with a review of the literature on its diverse histopathologic presentation. Case Rep Dent 2013;1-6  Back to cited text no. 10
    
11.
Eshghyar N, Jalayer-Nadery N, Ashery R. Calcifying odontogenic cyst: An analysis of thirty-six cases. Acta Medica Iranica 2006;44:59-62.  Back to cited text no. 11
    
12.
Manveen JK, Subramanyam RV, Simmerpreet SV, Ramandeep NS. Calcifying cystic odontogenic tumour mimicking as a residual cyst. J Clin Diagn Res 2010;4:2979-83.  Back to cited text no. 12
    
13.
Philipsen HP, Reichart PA. Calcifying epithelial odontogenic tumour: Biological profile based on 181 cases from the literature. Oral Oncol 2000;36:17-26.  Back to cited text no. 13
    


    Figures

  [Figure 1], [Figure 2], [Figure 3]



 

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