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 Table of Contents  
CASE REPORT
Year : 2013  |  Volume : 4  |  Issue : 3  |  Page : 132-134

Keratocystic odontogenic tumor invading the left maxilla: A rare case report


1 Department of Pedodontics, Government Dental College & Hospital, Maharashtra, India
2 Department of Oral Diagnosis Medicine & Radiology, VSPM Dental College & Research Center, Nagpur, Maharashtra, India
3 Department of Oral Pathology & Microbiology, Government Dental College & Hospital, Maharashtra, India

Date of Web Publication20-Nov-2013

Correspondence Address:
Ritesh R Kalaskar
Plot No. 68, Banerjee Layout, Bhagwan Nagar Road, Nagpur, Maharashtra
India
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Source of Support: None, Conflict of Interest: None


DOI: 10.4103/0976-433X.121641

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  Abstract 

Keratocystic odontogenic tumor (KCOT) is an intraosseous neoplasm of odontogenic origin characterized by high recurrence rates. Commonly, observed in males in second and third decades of life. The aim is to discuss the treatment modalities of a rare case of KCOT in a 5-year-old female child. Considering the age of the patient KCOT was treated by enucleation and Carnoy's solution application to reduce the recurrence and complications associated with repeated surgeries.

Keywords: Carnoy′s solution, enucleation, keratocystic odontogenic tumor, marsupialization, maxilla


How to cite this article:
Kalaskar RR, Kalaskar AR, Pol CA, Ghige SK. Keratocystic odontogenic tumor invading the left maxilla: A rare case report. SRM J Res Dent Sci 2013;4:132-4

How to cite this URL:
Kalaskar RR, Kalaskar AR, Pol CA, Ghige SK. Keratocystic odontogenic tumor invading the left maxilla: A rare case report. SRM J Res Dent Sci [serial online] 2013 [cited 2022 Oct 3];4:132-4. Available from: https://www.srmjrds.in/text.asp?2013/4/3/132/121641


  Introduction Top


Odontogenic keratocyst (OKC) is a developmental cyst as described by Philipsen in 1956. [1] World Health Organization (WHO) now referred OKC as keratocystic odontogenic tumor (KCOT) and defined it as ''a benign uni-or multi-cystic, intraosseous tumor of odontogenic origin, with a characteristic lining of parakeratinized stratified squamous epithelium and potential for aggressive, infiltrative behavior. [2] KCOTs often tend to recur after treatment because of: (a) Thin, friable wall which is often difficult to enucleate from the bone in one piece and (b) presence of small satellite cysts within the fibrous wall. It tends to grow in the anteroposterior direction within the medullary cavity of the bone without causing obvious bone expansion due to delayed observation by the patients. It is more commonly observed in males. Posterior body-ramus, maxillary third molar and canines are the prime location for KCOT. Usually, they are associated with unerupted tooth, pain, paresthesia, purulent discharge and may resorb or displace teeth. [2],[3]

The management of the KCOT remains controversial. Treatments are generally classified as conservative and aggressive. Conservative treatment generally includes simple enucleation, with or without curettage or marsupialization. Aggressive treatment generally includes peripheral ostectomy, enucleation with Carnoy's solution application, cryotherapy or electrocautery and resection. KCOTs are mostly observed in second or third decade of life and hardly seen in children below 5 years of age. [4] Owing to these unique observations KCOTs still present an important problem to pediatric surgeons. Presented here is a rare case of KCOT in a 5-year-female child managed successfully by surgical enucleation and Carnoy's solution application.


  Case Report Top


A 5-year-old girl presented with the chief complaint of swelling on the left side of face with a 6-month history of a slowly enlarging swelling on the same region that caused facial asymmetry. The swelling was painless with no history of nasal discharge and visual disturbance. There was no history of trauma to the left side of the face. Extraoral examination revealed diffuse swelling of the left cheek. Intraorally, the swelling extended from 62 to 55 obliterating the buccal vestibule [Figure 1]. None of the primary teeth in the involved region were carious. A panoramic radiograph revealed a well-defined corticated radiolucent lesion measuring 5 cm × 5 cm in diameter, which extended from mesial aspect of unerupted 22 to mesial aspect of unerupted 26. Tooth bud of 23 and 24 were displaced apically. 25 were missing [Figure 2]. Fine-needle aspiration was done which yielded yellowish white suspension of keratin, which has an appearance of pus, but without an offensive smell.
Figure 1: Intraoral photograph showing swelling extended from 62 to 65

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Figure 2: Osteoprotegerin showing well-defined corticated radiolucent lesion extended from 22 to 26

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Based on the history (no history trauma to the left side of face), clinical examination (no carious lesion) and fine needle aspiration observation a pre-operative diagnosis of KCOT was made. Dentigerous cyst and unicystic ameloblastoma were considered in the differential diagnosis. Under local anesthesia (Lox 2%, Mumbai) maxillary left primary first molar was extracted and the tumor was surgically enucleated. Displaced maxillary left first premolar attached to the cyst wall was also removed. Copious irrigation of bony cavity was done with betadine solution and normal saline. Carnoy's solution was applied in the bony cavity for 5 min to prevent the recurrence. Bony cavity was then packed with glycerin and betadine soaked ribbon gauze and horizontal matrix sutures with mersilk (Ethicon, Johnson and Johnson) were given. Post-operatively, the patient was assessed for parasthesia of the lip or cheek. The specimen sent for histopathological examination. The hematoxylin and eosin stained section showed uniform squamous epithelial lining, five to eight cells thick with fairly flat base. The epithelium demonstrated a well-developed basal layer of palisaded, cuboidal or columnar cells with polarized, hyperchromatic nuclei [Figure 3]. The histopathological examination confirmed the diagnosis of KCOT. In the subsequent visits removable space maintainer was given to maintain the space created by premature loss of maxillary left primary first molar. Considering the risk of recurrence, a long term follow-up period was planned. After 1 year follow-up, panoramic radiograph showed deposition of new bone in the bony cavity and no signs of recurrence [Figure 4].
Figure 3: Photomicrograph of keratocystic odontogenic tumor showing squamous epithelial lining, five to eight cells thick with fairly flat base

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Figure 4: Follow-up panoramic radiograph after 1 year

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  Discussion Top


KCOTs comprise approximately 11% of all cysts of the jaw. [4] It is one of the most aggressive odontogenic tumor having relatively high recurrence rate. It has even been reported to penetrate the base of the skull. KCOTs is commonly observed in males in second or third decades of life, especially involving posterior body and ramus region. [3] However, in the present case KCOT was observe in a 5-year-old female child involving posterior maxilla.

Literature suggests an association between high recurrence rate and KCOTs. Similarly, there is also an association between recurrence rate of KCOTs and treatment modality. [5],[6] As per recent reclassification of WHO, KCOTs is an aggressive neoplasm which warrants aggressive strategy such as peripheral ostectomy, enucleation with Carnoy's solution or en block resection. Most of the world-wide research showed that marsupialization has the highest recurrence rate followed by enucleation, enucleation and Carnoy's solution application and surgical resection of jaws. [4],[5],[6] Study conducted by Zecha et al. [6] showed that 40% of the marsupialized KCOTs recurred in a mean follow-up period of 58 months. Hyun et al. [7] reported a case of KCOT, which showed recurrence after first marsupialization but was uneventful for a period of 3 years and 9 months after second marsupialization. On the contrary one of the recent study suggested that marsupialization alone has promising results. However, the sample size and follow-up period of this study is too small to draw a definitive conclusion. [8] Literature review suggests that enucleation technique with the use of Carnoy's solution provide less recurrence rate than any other treatment modality except resection. [9] Resection on the other side is considered to be an aggressive and an extreme form of therapy for young children. Therefore enucleation and Carnoy's solution application seems to be less aggressive, hence can be considered as the choice of treatment for large KCOTs particularly in young children. This type of aggressive treatment modality has two main advantages: (1) Reduces the risk of recurrence; and (2) reduces the risk of trauma associated with separate surgeries.

Literature review revealed mixed information regarding age of patient and recurrence of KCOTs. Some reports have demonstrated that younger patients had significantly higher recurrence rates, whereas others have shown a significantly higher recurrence rate in older age. [10],[11] A study conducted by Titinchi and Nortje [3] showed that patients in their second and eighth decades of life experienced significantly higher recurrence than patients of other age groups. The association between recurrence rate and size of the lesion is controversial. Madras and Lapointe [12] reported an association between size of the tumor and recurrence: that even smaller tumor is associated with recurrence, whereas Forssell [11] reported no such association. Site of the KCOTs can be another factor that may have an association with recurrence. Literature search revealed both maxillary and mandibular posterior region are prone for recurrence. [2],[13] The possible reasons for recurrence are; (a) reduced accessibility to the area (b) involvement of adjacent structures like maxillary sinus.

Recurrence in preschool age exposed the child to recurrent surgeries and associated complication such as psychological trauma, fear of surgeries and dental treatment and associated social problems. Therefore in the present case considering the age of the patient and size of the lesion we preferred enucleation and Carnoy's solution application over marsupialization. We also provided space maintainer to preserve the space for future fixed prosthesis such as fixed partial denture or implant.


  Conclusion Top


Surgical enucleation and Carnoy's solution application is the choice of treatment for highly aggressive and recurrent KCOT. Age factor should be considered in the treatment of KCOT. The patients should be on clinical and radiographic follow-up for indefinite prognosis regardless of the treatment protocol.

 
  References Top

1.Philipsen HP. On keratocyst in the jaws. Tandlaegebladet 1956; 60: 963-80.  Back to cited text no. 1
    
2.Barnes L, Eveson JW, Reichart P, Sidransky D, editors. Pathology and Genetics of Head and Neck Tumours. Lyon: IARC Press; 2005. WHO classification of tumours series.  Back to cited text no. 2
    
3.Titinchi F, Nortje CJ. Keratocystic odontogenic tumor: A recurrence analysis of clinical and radiographic parameters. Oral Surg Oral Med Oral Pathol Oral Radiol 2012;114:136-42.  Back to cited text no. 3
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4.Neville BW, Damm DD, Allen CM, Bouquot JE. Oral and Maxillofacial Pathology. 2 nd ed. Philadelphia: Saunders; 2002. p. 595.  Back to cited text no. 4
    
5.Morgan TA, Burton CC, Qian F. A retrospective review of treatment of the odontogenic keratocyst. J Oral Maxillofac Surg 2005;63:635-9.  Back to cited text no. 5
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6.Zecha JA, Mendes RA, Lindeboom VB, van der Waal I. Recurrence rate of keratocystic odontogenic tumor after conservative surgical treatment without adjunctive therapies - A 35-year single institution experience. Oral Oncol 2010;46:740-2.  Back to cited text no. 6
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7.Hyun HK, Hong SD, Kim JW. Recurrent keratocystic odontogenic tumor in the mandible: A case report and literature review. Oral Surg Oral Med Oral Pathol Oral Radiol Endod 2009;108:e7-10.  Back to cited text no. 7
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8.Tabrizi R, Özkan BT, Dehgani A, Langner NJ. Marsupialization as a treatment option for the odontogenic keratocyst. J Craniofac Surg 2012;23:e459-61.  Back to cited text no. 8
    
9.Johnson NR, Batstone MD, Savage NW. Management and recurrence of keratocystic odontogenic tumor: a systematic review. Oral Surg Oral Med Oral Pathol Oral Radiol 2012;6:126-32.  Back to cited text no. 9
    
10.Myoung H, Hong SP, Hong SD, Lee JI, Lim CY, Choung PH, et al. Odontogenic keratocyst: Review of 256 cases for recurrence and clinicopathologic parameters. Oral Surg Oral Med Oral Pathol Oral Radiol Endod 2001;91:328-33.  Back to cited text no. 10
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11.Forssell K. The primordial cyst. A clinical and radiographic study. Proc Finn Dent Soc 1980;76:129-74.  Back to cited text no. 11
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12.Madras J, Lapointe H. Keratocystic odontogenic tumour: Reclassification of the odontogenic keratocyst from cyst to tumour. Tex Dent J 2008;125:446-54.  Back to cited text no. 12
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13.Habibi A, Saghravanian N, Habibi M, Mellati E, Habibi M. Keratocystic odontogenic tumor: A 10-year retrospective study of 83 cases in an Iranian population. J Oral Sci 2007;49:229-35.  Back to cited text no. 13
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  [Figure 1], [Figure 2], [Figure 3], [Figure 4]



 

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