Table of Contents  
CASE REPORT
Year : 2016  |  Volume : 9  |  Issue : 3  |  Page : 403-407  

Keratocystic odontogenic tumor of mandible


Department of Oral Medicine and Radiology, Vyas Dental College and Hospital, Jodhpur, Rajasthan, India

Date of Web Publication17-May-2016

Correspondence Address:
Ankita Bohra
Vyas Dental College and Hospital, Jodhpur - 341 008, Rajasthan
India
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Source of Support: None, Conflict of Interest: None


DOI: 10.4103/0975-2870.182524

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  Abstract 

Keratocystic odontogenic tumor is considered to be a benign cystic neoplasia of jaw bone with a higher rate of recurrence. It is noted to be third most common odontogenic cyst after radicular and dentigerous cyst. Most common location is posterior mandible and ascending ramus. A case with odontogenic keratocyst of the right posterior mandible is presented with proper clinical and radiographic examinations along with histopathological investigation. Cystic lesion was surgically resected with iliac crest bone graft replacement and reconstruction plate placement. Follow-up of 1 year is completed with repeated radiographic examinations in 4 months interval with no evidence of recurrence.

Keywords: Cystic neoplasm, keratocystic odontogenic tumor, odontogenic cyst, odontogenic keratocyst


How to cite this article:
Bohra A, Udawat V, Bhemappa A, Arya S. Keratocystic odontogenic tumor of mandible. Med J DY Patil Univ 2016;9:403-7

How to cite this URL:
Bohra A, Udawat V, Bhemappa A, Arya S. Keratocystic odontogenic tumor of mandible. Med J DY Patil Univ [serial online] 2016 [cited 2020 Nov 25];9:403-7. Available from: https://www.mjdrdypu.org/text.asp?2016/9/3/403/182524


  Introduction Top


Keratocystic odontogenic tumor (KCOT) is a benign cystic neoplasia of jaw bone. It has unique histopathological and clinical behavior because of its higher rate of recurrence and capacity of epithelial turnover as compared to other odontogenic cysts. It was first described by Philipsen in 1956 as (OKC). [1] Due to its neoplastic potential, OKC are considered to be benign cystic neoplasms rather than cysts. It has been designated as "KCOT" by World Health Organization (WHO) classification of head and neck tumors in the year 2005. [2] Presenting here a case report of KCOT of the mandible with a review of literature.


  Case Report Top


A 45-year-old male was referred to Department of Oral medicine and Radiology with a complaint of pain and swelling in lower right back jaw since 2-3 years [Figure 1] and [Figure 2]. Pain was continuous and dull in nature. There was no previous history of any trauma or any other surgical procedure. Medical history was unremarkable. The patient gave no history of hazardous habit. On local examination, slight facial asymmetry was present because of a diffuse swelling was presented over the right mandibular angle, approximately 6 cm × 5 cm in its anteroposterior dimension and 4 cm × 3 cm superioinferiorly. Swelling was nontender and firm in consistency. Skin over the swelling was normal. The right mandibular lymph node was palpable, mobile, and slightly tender. On intraoral examination, mouth opening was normal, mild vestibular tenderness was present with respect to the posterior right buccal vestibule [Figure 3] and [Figure 4]. Radiographic examination was suggested (intraoral peri-apical radiograph [IOPA], mandibular occlusal, orthopantamogram, computed tomography [CT] scan). IOPA of the right posterior mandible with respect to 46, 47 revealed a well-defined radiolucency present distal to the distal root of 47, with slightly scalloped borders [Figure 5]. Occlusal radiograph revealed no bucco-lingual expansion of cortical plates in favor of OKC [Figure 6]. On panoramic radiograph an apparently multilocular unilateral radiolucency was present on right mandible involving mandibular angle ramus on the right side 6 cm × 4 cm in greatest in anteroposterior dimension and 3 cm × 3 cm superioinferiorly with incomplete septa present within internal radiolucent lumen giving multilocular appearance [Figure 7]. CT scan was performed to measure the accurate extend of the lesion within the bone. CT examination of mandible, a multilocular unilateral expansile lesion was seen in right posterior mandibular angle and ascending right ramus of the mandible. About 29 mm anteroposterior, 20 mm transversely and 29 mm in vertical dimensions size of Lesion caused a breach of the mandibular cortex [Figure 8] and [Figure 9]. Radiographic diagnosis KCOT of the right mandible was given. Aspiration of the cystic lumen was taken which consisted of cheesy material with a clear fluid. Incisional biopsy was done. Histopathologic examination showed cyst wall consisted of 7-8 cells thick keratinized stratified squamous epithelium and noninflammatory fibrous connective tissue wall along with small satellite islands of odontogenic epithelium and keratinaceous debris [Figure 10]. Based on clinical and histo-pathological investigations, a final diagnosis of a KCOT. Surgical segmental resection of the right mandible was performed with removal of complete cystic lining with surrounding soft tissue with the use of Carnoy's solution, to minimize the chances of recurrences followed by iliac crest graft placement held in place by reconstruction plate placement. Post-operatively patient managed with intravenous antibiotics, analgesics and other supportive measures. The patient is under adequate follow-up from past 1 year with repeated panoramic radiographs taken after 3 months duration and showed no signs of recurrence of the lesion [Figure 11].
Figure 1: Front profile

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Figure 2: Extraoral swelling

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Figure 3: Intraoral view

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Figure 4: Right buccal vestibule

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Figure 5: Intraoral periapical radiograph showing diffuse radiolucency distal to 47

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Figure 6: Occlusal radiograph

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Figure 7: Orthopantamogram showing a unilateral diffuse multilocular radiolucency in right mandibular ramus

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Figure 8: Computed tomography three-dimensional reconstruction

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Figure 9: Three-dimensional reconstructed image

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Figure 10: Histopathologic image confirming odontogenic keratocyst

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Figure 11: Hemi resection of mandible followed by titanium reconstruction plate

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  Discussion Top


KCOT is defined 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." It arises from the dental lamina or basal cells of the oral epithelium.

WHO in 2005 included OKC under a neoplasm. Most commonly occur in third and fifth decade. There is slight male predilection. [3] Mandibular occurrences are more as compared to the maxilla, with a marked tendency to involve posterior body and ascending mandibular ramus. [4] OKC tends to grow in anteroposterior direction within the medullary cavity without causing much of buccolingual bone expansion. Radiographically, a well-defined radiolucency with smooth corticated sometimes scalloped margins with internal septae within the medullary cavity gives it a multilocular appearance. [5],[6] The lumen may be cloudy because of keratin, and the CT number of luminal contents may be higher than the other cystic fluids. Sometimes, focal areas of perforation of cortical plates may be seen in plain radiographs and CT. Approximately, 20-45% of cases documented associated with unerupted tooth and can provide a diagnosis of a dentigerous cyst. [7],[8] OKC shows less instances of root resorption as compared to the dentigerous cyst. The histological finding shows thin uniform fibrous wall with non-inflammatory changes and small satellite cysts/island of odontogenic epithelium. [9],[10] The thickness of cystic stratified epithelium changes to 6-8 cells thick with a detachment of fibrous wall. The lumen shows parakeratotic epithelial cells giving a wavy corrugated appearance. [11],[12],[13],[14] Basal cell layer composed of palisaded arrangement of the cuboidal/columnar hyper chromatic epithelial cells. [15],[16],[17] High recurrence rate can be attributed to the satellite cysts that are retained during enucleation, thin, and fragile cystic wall that can be left behind. Carnoy's solution acts as a fixative agent and its use in odontogenic cysts and tumors shows a reduction in recurrence rate to 9%.

The differential diagnosis for KCOT includes ameloblastoma, central giant cell granuloma, odontogenic myxoma, calcifying epithelial odontogenic cyst, and dentigerous cyst. The tendency for multiplicity associated with a gene level disturbance of chromosome 9 as in nevoid basal cell carcinoma syndrome, also referred to as Gorlin-Goltz syndrome. It is s transmitted as an autosomal dominant trait.

Multiple OKCs are associated along with other manifestations such as including multiple basal cell carcinomas, benign dermal cysts, palmar-plantar pits; cranio-dentofacial anomalies, such as OKC, malocclusion, broad nasal bridge, and increased head circumference; skeletal anomalies, including frontal and parietal bossing and mandibular prognathism, as well as costal anomalies involving the rib and vertebrae; ophthalmologic abnormalities, including hypertelorism, congenital blindness, and strabismus; and neurologic anomalies, including calcifications of the falx cerebri, bony bridging of the sella turcica, and medulloblastoma.

Teaching point

OKC is to an important odontogenic tumor with neoplastic characteristics. Proper clinical and radiographic examination along with histopathological investigations should be performed. Due to its recurring nature, adequate follow-up is a must at least for 3-5 years of duration including clinical and radiographical check-ups.

Declaration of patient consent

Th e 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 eff orts 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.
Philipsen HP. Om keratocyster (kolesteatom) I kaekberne. Tandlaegegebladet 1956;60:963-81.  Back to cited text no. 1
    
2.
Cakur B, Miloglu O, Yolcu U, Göregen M, Gürsan N. Keratocystic odontogenic tumor invading the right maxillary sinus: A case report. J Oral Sci 2008;50:345-9.  Back to cited text no. 2
    
3.
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. 3
    
4.
Boffano P, Ruga E, Gallesio C. Keratocystic odontogenic tumor (odontogenic keratocyst): Preliminary retrospective review of epidemiologic, clinical, and radiologic features of 261 lesions from University of Turin. J Oral Maxillofac Surg 2010;68:2994-9.  Back to cited text no. 4
    
5.
Mendes RA, Carvalho JF, van der Waal I. Characterization and management of the keratocystic odontogenic tumor in relation to its histopathological and biological features. Oral Oncol 2010;46:219-25.  Back to cited text no. 5
    
6.
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. 6
    
7.
Gomes CC, Diniz MG, Gomez RS. Review of the molecular pathogenesis of the odontogenic keratocyst. Oral Oncol 2009;45:1011-4.  Back to cited text no. 7
    
8.
Almeida P Jr, Cardoso Lde C, Garcia IR Jr, Magro-Filho O, Luvizuto ER, Felipini RC. Conservative approach to the treatment of keratocystic odontogenic tumor. J Dent Child (Chic) 2010;77:135-9.  Back to cited text no. 8
    
9.
Shear M. The aggressive nature of the odontogenic keratocyst: Is it a benign cystic neoplasm? Part 1. Clinical and early experimental evidence of aggressive behaviour. Oral Oncol 2002;38:219-26.  Back to cited text no. 9
    
10.
Henley J, Summerlin DJ, Tomich C, Zhang S, Cheng L. Molecular evidence supporting the neoplastic nature of odontogenic keratocyst: A laser capture microdissection study of 15 cases. Histopathology 2005;47:582-6.  Back to cited text no. 10
    
11.
Agaram NP, Collins BM, Barnes L, Lomago D, Aldeeb D, Swalsky P, et al. Molecular analysis to demonstrate that odontogenic keratocysts are neoplastic. Arch Pathol Lab Med 2004;128:313-7.  Back to cited text no. 11
    
12.
DelBalso AM. An approach to the diagnostic imaging of jaw lesions, dental implants, and the temporomandibular joint. Radiol Clin North Am 1998;36:855-90, vi.  Back to cited text no. 12
    
13.
Som PM, Curtin HD. Chronic inflammatory sinonasal diseases including fungal infections. The role of imaging. Radiol Clin North Am 1993;31:33-44.  Back to cited text no. 13
    
14.
Kimonis VE, Goldstein AM, Pastakia B, Yang ML, Kase R, DiGiovanna JJ, et al. Clinical manifestations in 105 persons with nevoid basal cell carcinoma syndrome. Am J Med Genet 1997;69:299-308.  Back to cited text no. 14
    
15.
Ahn SG, Lim YS, Kim DK, Kim SG, Lee SH, Yoon JH. Nevoid basal cell carcinoma syndrome: A retrospective analysis of 33 affected Korean individuals. Int J Oral Maxillofac Surg 2004;33:458-62.  Back to cited text no. 15
    
16.
Zachariades N, Papanicolaou S, Triantafyllou D. Odontogenic keratocysts: Review of the literature and report of sixteen cases. J Oral Maxillofac Surg 1985;43:177-82.  Back to cited text no. 16
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17.
Gorlin RJ. Nevoid basal-cell carcinoma syndrome. Medicine (Baltimore) 1987;66:98-113.  Back to cited text no. 17
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    Figures

  [Figure 1], [Figure 2], [Figure 3], [Figure 4], [Figure 5], [Figure 6], [Figure 7], [Figure 8], [Figure 9], [Figure 10], [Figure 11]



 

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