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 Table of Contents  
CASE REPORT
Year : 2014  |  Volume : 26  |  Issue : 1  |  Page : 103-106

Keratocystic odontogenic tumor of the right mandibular condyle: A rare case


Department of Oral Medicine and Radiology, Narsinhbhai Patel Dental College and Hospital, Gujarat, India

Date of Submission15-Aug-2014
Date of Acceptance13-Aug-2014
Date of Web Publication26-Sep-2014

Correspondence Address:
Kamala Rawson
Department of Oral Medicine and Radiology, Narsinhbhai Patel Dental College and Hospital, Visnagar - 384 315, Gujarat
India
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Source of Support: None, Conflict of Interest: None


DOI: 10.4103/0972-1363.141874

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   Abstract 

Odontogenic keratocyst (OKC) was first described by Hans Philipsen in 1956. The World Health Organization (WHO) has designated OKC as a Keratocystic Odontogenic Tumor (KCOT). KCOT is defined as 'a benign uni- or multicystic, intraosseous tumor of odontogenic origin, with a characteristic lining of parakeratinized stratified squamous epithelium, with a potential for aggressive, infiltrative behavior'. Radiographically, most OKCs are unilocular, presenting a well-defined peripheral rim and a central cavity having satellite cysts. It is characterized by a thin fibrous capsule and a lining of keratinized stratified squamous epithelium, which is typically corrugated, usually about 6 to 10 cells in thickness, and generally without rete pegs. The present case report describes an unusual case of KCOT with minimal clinical presentation indicative of a tumor highlighting the potential difficulties in determining the diagnosis.

Keywords: Cone beam computed tomography (CBCT), head of the condyle, keratocystic odontogenic tumor, unilocular


How to cite this article:
Rawson K, Kallalli BN, Telkar S, Penumatcha MR. Keratocystic odontogenic tumor of the right mandibular condyle: A rare case . J Indian Acad Oral Med Radiol 2014;26:103-6

How to cite this URL:
Rawson K, Kallalli BN, Telkar S, Penumatcha MR. Keratocystic odontogenic tumor of the right mandibular condyle: A rare case . J Indian Acad Oral Med Radiol [serial online] 2014 [cited 2020 Jul 6];26:103-6. Available from: http://www.jiaomr.in/text.asp?2014/26/1/103/141874


   Introduction Top


Keratocystic odontogenic tumor (KCOT), previously called an odontogenic keratocyst (OKC), is a cystic lesion of odontogenic origin and is classified as a developmental cyst derived from the dental lamina. [1] Odontogenic keratocysts can arise sporadically or in association with the nevoid basal cell carcinoma syndrome (NBCCS) or the Gorlin Syndrome. The odontogenic keratocyst was first discovered by Philipsen in 1956. [2] The World Health Organization (WHO) defined keratocystic odontogenic tumor as, 'a benign uni- or multicystic, intraosseous tumor of odontogenic origin, with a characteristic lining of a parakeratinized stratified squamous epithelium, with a potentially aggressive, infiltrative behavior'. [3] Although a considerable predilection for the mandibular third molar area and ascending ramus exists, KCOTs also occur in the dentate area, both in the maxilla and mandible, presenting themselves as seemingly ordinary odontogenic cysts. An extension of the cavity up to the ramus and even into the neck of the condyle is characteristic. Radiologically there are four variants of the keratocystic odontogenic tumor. They are:

  1. Envelopmental,
  2. Replacement,
  3. Extraneous, and
  4. Collateral. [4]


A very rare case of a KCOT in the right mandibular condyle with a review of literature is described.


   Case Report Top


A 45-year-old male patient reported to the department of Oral Medicine and Radiology with a chief complaint of pain in the right side of the face, in front of the ear, since 15 days [Figure 1]. The pain was sudden in onset of a severe lancinating type, continuous in nature, radiating to the right ear and forehead. The pain aggravated on opening the mouth and was relieved temporarily by taking medication. No other associated symptoms were present. He gave a history of trauma 20 years ago for which he underwent treatment for facial fractures.
Figure 1: Photograph of the patient

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On extraoral examination, the right temporomandibular joint (TMJ) was tender to palpation. No clicking or popping sounds were appreciated. The joint movements were less palpable on the right side and there was deviation of the jaw to the affected side on opening. The intraoral findings were non-significant. On the basis of these findings a provisional diagnosis of right TMJ capsulitis was arrived at. The patient was subjected to further investigations.

The panoramic radiograph [Figure 2] and [Figure 3] revealed a solitary, well-defined, unilocular radiolucency in the right condylar head area, measuring 1 × 1 cm in diameter. The margins of the lesion were continuous, extending up to the posterior border of the head of the condyle. The center of the lesion was radiolucent. The superior articular surface of the condylar head was discontinuous and irregular.
Figure 2: Orthopantomograph of the patient

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Figure 3: TMJ tomography of the patient

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The CBCT scans of the coronal and sagittal sections revealed medial cortical plate perforation in the right condylar head [Figure 4].
Figure 4: Cone beam computed tomography images

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With these clinical and radiographic findings, a differential diagnosis of traumatic bone cyst, pseudocyst of the condyle, central hemangioma, metastatic bone tumors, and OKC were included.

The lesion was managed surgically. Excision of the lesion was performed under general anesthesia [Figure 5].
Figure 5: Excised specimen

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The histopathological examination revealed a parakeratinized stratified squamous epithelium which was six to eight layers thick. The basal cells exhibited columnar cells with palisaded arrangements. The nucleus appeared polarized and intensely stained. The surface of the cystic lining showed a corrugated appearance. The underlying connective tissue capsule appeared thin, with parallel arrangement of the collagen fibres. The features were suggestive of a KCOT [Figure 6].
Figure 6: Histopathological appearance

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   Discussion Top


Odontogenic keratocysts account for 3-11% of the total jaw cysts. KCOT occurring in the condyle is very rare. The peak age of occurrence of KCOT is in the second and third decades of life. However, increased incidence has also been found in the fifth decade, which reflects its bimodal presentation, which has been confirmed by Rachanis and Shear. [5] There are mixed reports present in the literature for gender predilection. According to Khan et al., the occurrence of OKC is more in the body of the mandible (60%), and in the condyle it is 3.3%. [3] Our present case is similar to their study. According to Filip Brzozowski et al., OKCs are more common in the mandibular ramus and body (15 cases), and in condyle they have found only one case. [2] The present case is similar to their study. The mandible is involved more commonly than the maxilla (77%). About half of KCOTs occur at the angle of the mandible, extending for variable distances into the ascending ramus and forward into the body. [6] According to Stoelinga, on a scan, KCOT presents as a relatively small unilocular cyst in the tooth region or a frequently large multilocular cyst in the posterior maxilla or angle-ramus region. [7] OKCs tend to grow in an anterior-posterior direction, within the medullary cavity of the bone, without causing any obvious bone expansion. [3]

The source for odontogenic epithelium has to be either remnants of dental lamina or cells of the basal layer of the oral mucosa. Origin from the former source is not possible because of the location. Therefore, the potential cells originate either from proliferation or as an offshoot from the lining of the basal lamina. [1]

Keratocystic odontogenic tumors are often asymptomatic, until the bone is expanded or the cyst gets infected. The expansion of the jaw is much less than would be expected from the radiographic extent of the cyst.

A keratocystic odontogenic tumor in the condyle often resembles a pseudocyst of the condyle or a traumatic bone cyst. In the present case, these have been excluded based on the CBCT findings of the medial cortical plate perforation, which is not commonly seen in the case of a pseudocyst.

A number of clinicians favor 'conservative' therapy, while others advocate more 'aggressive' forms of treatment. Meiselman et al. [8] suggested conservative therapies to include 'enucleation, curettage, and marsupialization'. Williams et al. [9] suggested aggressive treatment in addition to enucleation, and included curettage (mechanical, physical, and/or chemical) and/or resection with or without loss of jaw continuity. The treatment recommended for these lesions varies from most conservative measures, including marsupialization, with or without enucleation, to extensive procedures, including enucleation with adjuvant procedures, such as, application of Carnoy's solution or liquid nitrogen cryotherapy to procedures like resection, which carries the highest morbidity.

Histopathological features showed a parakeratinized stratified squamous epithelium, which was six to eight layers thick. The basal cells exhibited columnar cells with palisaded arrangements. The nucleus appeared polarized and intensely stained. The surface of the cystic lining showed a corrugated appearance. The underlying connective tissue capsule appeared thin, with parallel arrangement of the collagen fibres. The histological features were consistent with those described in literature. [10]

Recurrence associated with resection is 0% and with simple enucleation combined with adjunctive therapy, it is 1-8.7%, while enucleation alone has been reported to have a recurrence rate of 17-56%. [11] Stoelinga recommends excision of the overlying attached mucosa along with enucleation, and treatment of the bony defect with Carnoy's solution. [7]


   Conclusion Top


To conclude, KCOT of the head of the condyle is very rare. Only few cases have been reported so far; hence, the oral physicians should have a thorough knowledge of the clinical, radiographic, and histopathological coordination, for appropriate diagnosis of these lesions. Further follow up of the lesion is necessary, as KCOT has a high recurrence rate due to the presence of satellite cysts.

 
   References Top

1.Rai KK, Amarnath PU, Batra J, Ashok L, Shivakumar HR, Chatura KR. Keratocystic odontogenic tumor of mandibular condyle. Int J Dent Case Reports 2013;3:113-7.  Back to cited text no. 1
    
2.Brzozowski F, Wanyura H, Stopa Z, Kowalska K. Odontogenic keratocysts in the material of the Department of Craniomaxillofacial Surgery, Medical University of Warsaw. Czas Stomatol 2010;63:69-78.  Back to cited text no. 2
    
3.Khan M, Din QU, Rehman AU. Clinical and radiological behavior of sporadic odontogenic keratocyst- A study. Pak Oral Dental J 2009;29:197-200.  Back to cited text no. 3
    
4.Hemavathy S, Roy S. Follicular odontogenic keratocyst mimicking dentigerous cyst- Report of two cases. Arch Oral Sci Res 2011;1:100-3.  Back to cited text no. 4
    
5.Rachanis CC, Shear M. Age-standardized incidence rates of primordial cyst (keratocyst) on the Witwatersrand. Community Dent Oral Epidemiol 1978;6:296-9.  Back to cited text no. 5
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6.Shear M, Speight P. Cysts of the Oral and Maxillofacial Regions. 4 th ed. Oxford: Blackwell Munksgaard; 2007. p. 6-58.  Back to cited text no. 6
    
7.Stoelinga PJ. The treatment of odontogenic keratocysts by excision of the overlying, attached mucosa, enucleation, and treatment of the bony defect with carnoy solution. J Oral Maxillofac Surg 2005;63:1662-6.  Back to cited text no. 7
[PUBMED]    
8.Meiselman F. Surgical management of the odontogenic keratocyst: Conservative approach. J Oral Maxillofac Surg 1994;52:960-3.  Back to cited text no. 8
[PUBMED]    
9.WilliamsTP, Connor FA Jr. Surgical management of the odontogenic keratocyst: Aggressive approach. J Oral Maxillofac Surg 1994;52:964-6.  Back to cited text no. 9
    
10.Rajendran R, Sivapathasundaram B. Shafer's Textbook of Oral Pathology. 5 th ed. New Delhi: Elsevier; 2006. p. 365.  Back to cited text no. 10
    
11.Blanas N, Freund B, Schwartz M, Furst IM. Systematic review of the treatment and prognosis of the odontogenic keratocyst. Oral Surg Oral Med Oral Pathol Oral Radiol Endod 2000;90:553-8.  Back to cited text no. 11
    


    Figures

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



 

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