International Journal of Oral & Maxillofacial Pathology. 2012;3(3):18-21 ISSN 2231 – 2250

Available online at http://www.journalgateway.com or www.ijomp.org

Case Report

Central Calcifying Odontogenic Cyst mimicking Central Giant Cell Granuloma: A Unique

Case Report

Bharti Saraf, Minal Chaudhary, Shekhar Gupta, Rahul Patil

Abstract

Calcifying cystic odontogenic tumour first described by Gorlin in 1962, accounts for less than 2%
of all odontogenic tumours. This rare cystic odontogenic lesion, sometimes referred to as the
‘keratinizing and calcifying odontogenic cyst’, is characterized by the presence of ‘ghost’ epithelial cells and by its resemblance to the pilomatrixoma of the skin. In this report, we present a rare case of calcifying odontogenic cyst mimicking as a central giant cell granuloma, in a 30 year old female patient present in the anterior region of lower jaw crossing the midline. The lesion was surgically removed. After enucleation no recurrence has been recorded during the follow up period of three years. Hence correlation of clinical and radiological information with histological features is important in the diagnosis of odontogenic tumours and cysts.

Key Words: Calcifying;Odontogenic Cyst;Odontogenic Tumours;Jaw Cyst;Granuloma;Giant Cell

Reparative;Jaw Diseases.

Bharti Saraf, Minal Chaudhary, Shekhar Gupta, Rahul Patil. Central Calcifying Odontogenic Cyst mimicking Central Giant Cell Granuloma: A Unique Case Report. International Journal of Oral and Maxillofacial Pathology; 2012:3(3):18-21. ©International Journal of Oral and Maxillofacial Pathology. Published by Publishing Division, Celesta Software Private Limited. All Rights Reserved.

Received on: 12/08/2011 Accepted on: 02/02/2012

Introduction

Calcifying odontogenic cyst (COC) was first categorized as distinct entity by Gorlin et al., in 1962 and was named after him since then. According to Shear it account for 1% of the jaw cysts. Once it was thought to be a cutaneous counter part of benign calcifying
epithelioma of malherb described in 1980.1
The COC is an unusual and unique lesion with characteristics of solid neoplasm and of a cyst. It shows considerable amount of histopathological diversity with variable clinical behavior, such as cystic, neoplastic and infiltrating malignant behavior. There may be variants of COC according to clinical, histopathological and radiological characteristics. There proper characterization of cases is needed for better understanding of pathogenesis of each variant.
The central giant cell granuloma (CGCG) of the jaws account for approximately 7% of all benign tumors of the jaws.2,3 These lesions
mainly occur in young adults with a predilection for females.3-6 The lesions must be differentiated from a variety of the jaw
lesions such as cysts, odontogenic tumors, fibroosseus lesions, vascular malformations and even malignancies.5,2 Radiographically it is a unilocular or multilocular radiolucency.2,7
As this is a slow growing lesion, borders are usually well defined. Teeth displacement,
root and lamina dura resorption of the teeth may also be observed.6,8

Case Report

A 30 year old female reported to Out Patient Department with chief complaint of an asymptomatic swelling in the lower jaw. The swelling was present since three months and was slowly enlarging to the present size of 6 x 3 cm. On extraoral evaluation an asymmetry involving lower right and left face region extending from right corner of mouth to two centimeter behind left corner of mouth was present. Borders of swelling were indistinct and variable in consistency on palpation. Consistency was firm on periphery and fluctuant at the center. Paraesthesia was present in the region of swelling. Intraoral examination revealed buccal cortical expansion extending from tooth #43 to #36 region crossing the midline. Mucosa over the swelling was intact. On palpation swelling was non tender and cystic. Mobility was present with tooth #42,
#41, #31, #32, #33, #34 and #35. Displacement of involved teeth was also noted.
Radiographic examination showed a well- defined unilocular radiolucency extending anteroposteriorly from distal of tooth #42 to mesial of #36. Superio-inferiorly the lesion extends from periapical region of involved teeth to lower border of mandible. There was

©2012 International Journal of Oral and Maxillofacial Pathology. Published by Publishing Division, Celesta Software Private Limited. All Rig hts Reserved

ISSN 2231 – 2250 Central Calcifying Odontogenic Cyst mimicking Central... 19

external root resorption of seven teeth from tooth #42 till the tooth #35 in apical 1/3rd area (fig 1c). Based on the clinical presentation and radiographic appearance
differential diagnosis of central giant cell granuloma, unicystic ameloblastoma and aneurysmal bone cyst was considered.
Before going for surgery 16 ml of cystic aspirate was drained with wide board needle using the thinnest part present on the buccal cortical plate. Cytological smear of the cystic aspirate was prepared and subjected to microscopic examination. It showed numerous pus cells, RBC’s and few cholesterol crystals which were non- conclusive. Full thickness envelop flap when raised showed loss of buccal cortical plate at the center of the lesion and thinning at the periphery. A well-defined cystic lesion was enucleated in toto and well separated from the surrounding bone. Inferior alveolar nerve was sacrificed, and then the whole specimen was subjected to histopathological examination.
On histopathological examination the microscopic features include a fibrous capsule with a stratified lining of odontogenic epithelium. The basal layer was made up of ameloblast-like columnar or cuboidal polarized cells of four to ten cell thickness over-lined by a loosely arranged spindle or stellate shaped odontogenic epithelial cells bearing similarity to stellate reticulum of the enamel organ. There exists varying number of epithelial cells devoid of nuclei which are eosinophilic and retain their basic cell outline suggestive of ghost cells (fig 1d). These ghost cells show calcification at some places and lose their cellular outline to firm sheet like area of calcified keratin. Calcification and dentinoid material was found. Ghost cells and dentinoid were very well demonstrated by the special stains like Mallory and Van Geison Stain. Accentuated ghost cells appear orange colored in Mallory stain and yellow colored in Van Geison stain (fig 1e & f).

Discussion

The age ranges from 1 to 82 years with peak incidence in the 2nd decade. In an observation of 215 lesions Bruchner et al., have drawn attention to bimodal age
distribution in support of their contention that
two different entities may be involved with second peak in sixth / seventh decade.9,10
The lesion has no sex predilection and is equally distributed between maxilla and
mandible.11 Radiographically majority of the lesions present in an unilocular form with well-defined margin,9,12 while in 5 - 13% of the cases they are multilocular. They have
scattered irregular sized calcification producing a variable range of opacities [salt and pepper type of patterns].12 They may be associated with tooth like densities in 50% of the cases and one-third of the cases show
association with unerupted tooth, most often a canine.13
The central COC presents as an asymptomatic hard swelling of the jaws that produces expansion rather than erosion of
bone. Pain indicates secondary infection.14-16
Early lesions are usually detected following routine radiographic examination and they are often associated with an unerupted tooth.1,17 The peripheral COC presents on the gingiva as a non-specific well-
circumscribed sessile or pedunculated mass
with a smooth surface. They resemble other epulis, such as gingival fibromas or peripheral giant cell granulomas.15 The definitive diagnosis of COC is made
histologically, due to the lesion's lack of
characteristic clinical and radiological features, as well as its variable biological behaviour.17
According to Buchner's classification16 this lesion would be classified as a central multicystic COC (type B 1a). The classification of Praetorius does not recognize multicystic lesions as a separate entity. The presence of radiopacities could suggest a diagnosis of an adenomatoid odontogenic tumour, a calcifying epithelial odontogenic tumour or an ameloblastic fibro-
odontoma.18 Nagao and colleagues19
reported in a series of 23 cases only three cases of multilocular lesions, which converts to a ratio of 7.6:1. In 33% of cases COCs are associated with unerupted teeth.
Classification of calcifying odontogenic cysts as proposed by Praetorius et al.20
1 a: Simple unicystic type with little or no dentinoid
1 b: Odontoma producing type
1 c: Ameloblasomatous proliferating type
2: Neoplastic variant (dentinogenic `ghost' cell tumor)
Classification of calcifying odontogenic cysts as proposed by Buchner16
A. Peripheral (extraosseous) COC
1. Cystic variant
2. Neoplastic (solid) variant

20 Bharti Saraf et al., ISSN 2231 - 2250

B. Central (intraosseous) COC
1. Cystic variant
i. Simple (unicystic or multicystic)
ii. Associated with an odontoma
iii. Associated with odontogenic tumours (other than odontoma)
iv. Other variants (such as clear cell variant, pigmented variant)
2. Neoplastic (solid) variant
3. Malignant COC
Clinically the lesion was present in lower anterior region crossing the midline as an
asymptomatic swelling; age of presentation was 3rd decade in female. The lesion was unilocular with well-defined borders without any radiopacities in the cystic lumen. All
these findings were in favor of central giant cell granuloma, unicystic ameloblastoma both of which are aggressive lesions and show definite degree of recurrence. After histopathological examination it came out to be calcifying odontogenic cyst which is a benign neoplasm and do not show recurrence.

Figure 1: Extraoral swelling in lower left chin region (a) with Intraoral buccal expansion (b) and drifting of teeth in the region of swelling and orthopantomograph showing radiolucency crossing the midline (c). The photomicrograph under low power view showing Ghost Cells in hematoxilin & eosin stain (d), Mallory Stain (e) and Van Geison Stain (f).

The clinical significance of this case is that clinical and radiographical observations appeared to be simple, unambiguous and obvious. However, the histopathological observations showed that it was a rare
odontogenic lesion. Hence, this case report emphasizes the role of biopsy in the diagnosis of such deceptive clinical presentations. This also proves the importance of microscopic examination of

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each and every case before going for the treatment which may change the final diagnosis of the patient and hence the treatment plan and the final outcome of the treatment.

Author Affiliations

1. Dr.Bharti Saraf, Senior Lecturer, Prabhu Dayal Memorial Dental College, Research Institute & Hospital, Aurangabad, 2. Dr.Minal Chaudhary, Professor and Head, Sharad Pawar Dental College & Hospital, Wardha, 3. Dr.Rahul Patil, Senior Lecturer, Department of Oral Pathology, Mahatma Gandhi Vidya Mandir’s KBH Dental College, Nasik, 4. Dr.Shekhar Gupta, Senior Lecturer, Department of Prosthodontics, Prabhu Dayal Memorial Dental College Research Institute & Hospital, Aurangabad, India.

Acknowledgement

We would like to thank all the staff members from

Department of Oral Pathology for their support.

References

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Corresponding Author

Dr Bharti Saraf,

C/261, Prashant Vihar, Rohini, Sector 14, New Delhi – 110085, India.

E-mail: drbhartisaraf@gmail.com

Source of Support: Nil, Conflict of Interest: None Declared


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