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Table of Contents   
CASE REPORT  
Year : 2011  |  Volume : 22  |  Issue : 1  |  Page : 181
Massive keratocystic odontogenic tumor of mandible: A case report and review of literature


1 Department of Oral and Maxillofacial Surgery, M R Ambedkar Dental College and Hospital, Bangalore, India
2 Department of Periodontics, M R Ambedkar Dental College and Hospital, Bangalore, India

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Date of Submission28-Dec-2009
Date of Decision06-Jul-2010
Date of Acceptance10-Nov-2010
Date of Web Publication25-Apr-2011
 

   Abstract 

Keratocystic odontogenic tumor (KCOT), also known as odontogenic keratocysts, as defined by World Health Organization (WHO), are known for their peculiar behavior, varied origin, debated development, unique tendency to recur, and disputed treatment modalities. We present a case of KCOT involving symphysis menti, right and left halves of the body of mandible in an 11-year-old girl treated with enucleation and open dressing (bismuth, iodoform, paraffin paste) with long-term follow-up.

Keywords: Anterior mandible, bismuth iodoform paraffin paste, enucleation and open dressing, keratocystic odontogenic tumor

How to cite this article:
Rajkumar G C, Hemalatha M, Shashikala R, Sonal P. Massive keratocystic odontogenic tumor of mandible: A case report and review of literature. Indian J Dent Res 2011;22:181

How to cite this URL:
Rajkumar G C, Hemalatha M, Shashikala R, Sonal P. Massive keratocystic odontogenic tumor of mandible: A case report and review of literature. Indian J Dent Res [serial online] 2011 [cited 2019 Aug 18];22:181. Available from: http://www.ijdr.in/text.asp?2011/22/1/181/80000
Odontogenic keratocyst (OKC) has been a subject of great interest since Philipsen introduced the term in 1956. The OKC was designated by the World Health Organization (WHO) in 2005 as a keratocystic odontogenic tumor (KCOT) [1] and is defined as "a benign uni or multicystic, intraosseous tumor of odontogenic origin, with a characteristic lining of parakeratinized stratified squamous epithelium and potential for aggressive, infiltrative behavior." WHO recommends the term "keratocystic odontogenic tumor" as it better reflects its neoplastic nature. Although there is considerable predilection for mandibular 3 rd molar region and ramus, anterior mandible is an uncommon site with the lesion crossing the midline, this being an unique occurrence. [2]


   Case Report Top


We present a case report of an 11-year-old female patient who visited the Department of Oral and Maxillofacial Surgery, M.R. Ambedkar Dental College and Hospital, Bangalore, India, for correction of malalignment of teeth. The patient was totally unaware of the lesion developing in her lower jaw. It was an accidental finding made when orthopantomogram (OPG) was taken for orthodontic purpose. Past medical and surgical history was non-contributory.

On extraoral examination, mandible was slightly prognathic with mild asymmetry of the face on the right side. Intraoral examination revealed missing right and left mandibular primary lateral incisors and mobile anterior teeth. Two mandibular permanent incisors were in erupting stage and slightly mobile. A single swelling was seen, which was non-tender, hard in consistency and diffuse, extending from right to left first permanent molar region, with expansion of buccal and lingual cortex and obliteration of buccal and labial vestibules [Figure 1]. The overlying mucosa was normal.
Figure 1: Intraoral photograph showing labial and buccal cortical expansion and obliteration of vestibules

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Panoramic view revealed unilocular, large radiolucency extending from mesial surface of 36 to distal surface of 46 regions with thinning of inferiorborder of the mandible. Multiple teeth in various stages of root formation were seen to be floating and displaced within radiolucency. Most of the teeth were displaced to the lower border of mandible. Root resorption was observed in all mandibular deciduous teeth. Inferior alveolar canal was not very evident with the possibility of displacement to inferior border of mandible [Figure 2]. Occlusal radiograph revealed bicortical expansion of the lower jaw with no root resorption of permanent mandibular incisors [Figure 3]. Computed tomography (CT) scan revealed large, expansile, lytic lesion involving symphysis menti, right and left halves of body measuring about 7.7 × 3.9 × 3.4 cm [Figure 4], [Figure 5] and [Figure 6]. Three-dimensional (3-D) CT scan revealed perforation of the right buccal cortex [Figure 7]. Hematological investigation was normal. Aspiration, incisional biopsy and histological examination confirmed parakeratotic type of OKC.
Figure 2: Panoramic view showing large radiolucent lesion with multiple displaced and unerupted teeth with paper thin lower border of mandible

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Figure 3: Occlusal radiograph showing bicortical expansion

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Figure 4: Axial CT showing extent of the lesion

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Figure 5: Axial CT showing right buccal cortical perforation

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Figure 6: Coronal CT showing expansile, lytic lesion with displaced teeth

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Figure 7: 3-D CT reconstruction showing marked expansion of the labial and buccal cortical plates extending from right to left permanent first molar

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We performed enucleation and curettage under general anesthesia. Two separate vestibular incisions were placed from second primary molar to lateral incisor region sparing symphysis menti region. The main reason behind this flap design was to maintain the contour and continuity of chin as well as maintaining esthetics at the same time. Cyst was enucleated through the bony windows; all the teeth within cystic cavity were removed [Figure 8]. After achieving hemostasis, the defect was filled by betadine soaked ribbon gauge. Postoperative antibiotics and analgesics were prescribed. Follow-up dressing was done with bismuth iodoform paraffin paste every alternate day for 2 months and later once a week. Acrylic prosthesis was given to the patient 9 months after surgery. Excellent healing was achieved after 1 year of follow-up. Size of the bony defect has been reduced to approximately 90% of original size both clinically and radiographically [Figure 9] and [Figure 10].
Figure 8: Enucleated cystic lining along with the teeth

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Figure 9: Panoramic view showing significant amount of bone formation at 9-month follow-up

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Figure 10: One-year postoperative occlusal radiograph showing significant amount of bone healing

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   Discussion Top


OKCs are relatively common developmental odontogenic cysts and account for 10-12% of all jaw cysts, with two peaks around the ages of 30 and 60, and seem to be more frequent in males (M:F = 2:1). Approximately, three-quarters of all OKCs occur in the body of the mandible, most commonly in the molar region and vertical ramus. An OKC usually occurs as a single lesion. Multiple lesions are associated with the nevoid basal cell syndrome (Gorlin-Goltz syndrome). Unlike other odontogenic cysts, OKCs have a high recurrence rate, reportedly ranging from 13 to 60%. In approximately 50% of patients, the lesion is asymptomatic. In others, pain, swelling, expansion, drainage, teeth mobility and bone perforation can be seen. On histopathologic examination, an OKC has a fibrous wall lined by keratinized stratified squamous epithelium, usually about six to eight cells in thickness and generally without rete pegs. [2] We present a parakeratotic type of OKC in this report. The luminal content can have different consistencies described as a "straw-colored fluid," "thick pus-like" material or a caseous, thick, cheesy, milk white mass. [3] The parakeratotic type is the most frequent (80%) and has a more aggressive clinical presentation than the orthokeratinized variant. [3]

OKCs have a tendency for intraosseous growth, more often in a longitudinal than in a transverse direction (minimal expansion), as seen in this case, thereby replacing the bone marrow, rather than giving rise to periosteal bone formation, which would result in a bony swelling. Rapid growth does not allow enough time for the periosteum to lay down new bone. [2] The following radiographic features are characteristic of OKCs: 1) a distinctly corticated, often scalloped, border; 2) expansion, especially toward the lingual (medial) side and growth along the length of the mandibular bone; 3) displacement of developing teeth and/or separation or resorption of the roots of erupted teeth and extrusion of erupted teeth; and 4) a radiolucent lumen, and occasionally, a cloudy or milky appearance of the lumen on the panoramic radiograph. High attenuation within an expansile benign lesion of the mandible on nonenhanced CT scans, with no enhancement after contrast material injection, can indicate an OKC. The high attenuation suggests the presence of a dense proteinaceous material such as keratin. [3] In the selection of the most appropriate treatment for OKC, one must take into consideration both patient factors and lesion characteristics. Patient factors include age and general medical condition. Lesion characteristics include size, location, extent, recurrent lesion, presence of cortical perforation and/or involvement of soft tissue and adjacent structures.

Conservative treatment is "cyst-oriented," and thus includes enucleation, with or without curettage, or marsupialization. Its advantage is preservation of anatomical structures (including teeth), which is advocated because KCOTs commonly present in younger patients as in this case. One disadvantage of marsupialization is the compliance required of the patient for a prolonged period of time. Failure of the procedure to eradicate the lesion will require subsequent enucleation or resection. Enucleation has an advantage over marsupialization of providing a complete specimen for histopathologic examination. In lesion of the mandibular symphysis, body and third molar regions, enucleation and curettage appear to be the most common method of treatment. Lesion of the ascending ramus, if extensive in size, may be more difficult to successfully treat in this manner. [4]

In Browne's series, three different treatment methods were evaluated, which were marsupialization, enucleation and primary closure, and enucleation and open dressing. There was no correlation between treatment method and the rate of recurrence. [5]

Recurrence of an OKC is due to the nature of the lesion itself, namely, the presence of additional remnants of dental lamina, from which a cyst might develop, high mitotic activity in basal cell layer, friable capsule, and inaccessible sites preventing complete removal. [Table 1] shows series of cases treated by enucleation and recurrence rate. A recent study suggests that PTCH1 mutations, particularly those causing protein truncations, are associated with OKCs showing increased proliferative activity and thus relate to a phenotype of higher recurrent tendency. [9] The use of Carnoy's solution, cryosurgery and soft tissue excision gives a very low incidence of relapse. [7] It is, however, advisable not to leave the carnoy's solution for too long in contact with the exposed nerve. [2] Fisher et al. found that the rat mandible undergoes a significant reduction in mechanical strength at 8 weeks after cryosurgery [10] and because of difficulty in controlling the amount of liquid nitrogen applied to the cavity, the resultant necrosis and swelling can be unpredictable. [11] Considering these facts, the use of these agents was not appropriate in this case. The modified Brosch procedure represents a reasonable alternative to resection or marsupialization, when treating large OKC. Extraction of teeth affected by the lesion as well as generous removal of partially eroded bone and overlying soft tissues may contribute to lower recurrence rates. [12] The Multidisciplinary Sequential Treatment (MST) approach consists of decompression, scaling of the capsule wall of OKC, cutting and grinding of sclerotin of bone cavum, and cauterizing bone cavum by using carbolic acid. [13]
Table 1: Series of odondotogenic keratocyst treated by enucleation

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There continues to be controversy, especially regarding the management of the large keratocyst, with cortical perforation and extension into soft tissue. In case of cortical perforation, will there be residual lesion or daughter cysts adherent to soft tissue or periosteum at the time of enucleation? To eliminate this possibility, the overlying bone and mucosa were excised in the area of perforation of bone in this case.

Aggressive modalities have generally been recommended for nevoid basal cell carcinoma syndrome (NBCCS) cases, large KCOTs and recurrent lesions. [1] Recently, reclassification of this lesion from cyst to tumor advocates aggressive treatment. But resection causes esthetic and functional damage which, especially among the young patients, could give a poor quality of life. [14] So, respecting the delicate anatomical structure of the jaws and giving the patient a better quality of life, this case was treated conservatively.


   Conclusion Top


Unfortunately, there is no consensus on a uniform treatment plan and the recommended surgical managements vary from marsupialization and enucleation to en bloc resection. The type of treatment chosen depends on several factors like patient age, lesion location and size and whether the KCOT is primary or recurrent. In this case, putting into consideration the young age of the patient, subsequent growth of mandible and facial contour, treatment was performed with enucleation and open dressing. In any case, clinical and radiographic follow-up is mandatory for years after surgery because recurrence of this lesion may occur even years later. As research continues, treatment may become molecular in nature. This could eventually reduce or eliminate the need for aggressive methods to manage the lesions.


   Acknowledgment Top


We would like to thank Dr. Hemavathy for her support and contribution in preparing the manuscript.

 
   References Top

1.Madras J, Lapointe H. Keratocystic Odontogenic Tumour: Reclassification of the Odontogenic Keratocyst from Cyst to Tumour. J Can Dent Assoc 2008;74:165. Available from:http://www.cda-adc.ca/jcda/vol-74/issue-2/165.html,www.cda-adc.ca/jcda [Last accessed on Mar 2008].  Back to cited text no. 1
    
2.Stoelinga PJ. Long term follow up on keratocysts treated according to defined protocol. Int J Oral Maxillofac Surg 2001;30:14-25.   Back to cited text no. 2
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3.Yoshiura K, Higuchi Y, Ariji Y, Shinohara M, Yuasa K, Nakayama E, et al. Increased attenuation in odontogenic keratocysts with computed tomography: A new finding. Dentomaxillofac Radiol 1994;23:138-42.  Back to cited text no. 3
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4.Giuliani M, Grossi GB, Lajolo C, Bisceglia M, Herb KE. et al. Conservative management of a large odontogenic keratocyst: Report of a case and review of literature. J Oral Maxillofac Surg 2006;64:308-16.  Back to cited text no. 4
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5.Browne RM. The odontogenic keratocyst: Clinical aspects. Br Dent J 1970;128:225-31.  Back to cited text no. 5
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6.Brannon RB. The odontogenic keratocyst-a clinicopathologic study of 312 cases. Part 1. Clinical features. Oral Surg Oral Med Oral Pathol 1976;42:54-72.  Back to cited text no. 6
    
7.Voorsmith RA, Stoelinga PJ, Van Haelst UJ. The Management of Keratocysts. J MaxilloFac Surg 1981;9:228-36.   Back to cited text no. 7
    
8.Kuroyanagi N, Sakuma H, Miyabe S, Machida J, Kaetsu A, Yokoi M, et al. Prognostic factors for keratocystic odontogenic tumor (odontogenic keratocyst): Analysis of clinicopathologic and immunohistochemical findings in cysts treated by enucleation. J Oral Pathol Med 2009;38:386-92.  Back to cited text no. 8
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9.Pan S, Li TJ. PTCH1 mutations in odontogenic keratocysts: Are they related to epithelial cell proliferation? Oral Oncol 2009;45:861-5.  Back to cited text no. 9
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10.Fisher AD, Williams DF, Bradley PF. The effect of cryosurgery on the strength of bone. Br J Oral Surg 1977;15:215-22.  Back to cited text no. 10
    
11.Schmidt BL, Pogrel MA. The use of enucleation and liquid nitrogen cryotherapy in the management of odontogenic keratocysts. J Oral Maxillofac Surg 2001;59:720-5.  Back to cited text no. 11
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12.Ephros H, Lee HY. Treatment of a large odontogenic keratocyst using the Brosch procedure. J Oral Maxillofac Surg 1991;49:871-4.   Back to cited text no. 12
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13.Sun J, Wu J, Zhou N, Meng N, Lin Y. A prospective study of jaw odontogenic keratocyst treated by multidisciplinary sequential treatment. Int J Oral Maxillofac Surg 2009;38:537.   Back to cited text no. 13
    
14.Myoung H, Hong SP, Hong SD, Lee JI, Lim CY, Choung PH, Lee JH, 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. 14
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Correspondence Address:
G C Rajkumar
Department of Oral and Maxillofacial Surgery, M R Ambedkar Dental College and Hospital, Bangalore
India
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Source of Support: None, Conflict of Interest: None


DOI: 10.4103/0970-9290.80000

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    Figures

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