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 Table of Contents  
CASE REPORT
Year : 2014  |  Volume : 26  |  Issue : 3  |  Page : 335-337

Keratocystic odontogenic tumor involving the maxillary antrum with displacement of the third molar: A rare case report


Department of Oral Medicine and Radiology, MNR Dental College and Hospital, Sangareddy, Telangana, India

Date of Submission26-Jun-2014
Date of Acceptance09-Nov-2014
Date of Web Publication19-Nov-2014

Correspondence Address:
Rashmi Shivakumar Doshetty
P.G. Student, Department of Oral Medicine and Radiology, MNR Dental College and Hospital, Sangareddy - 502 294, Telangana
India
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Source of Support: None, Conflict of Interest: None


DOI: 10.4103/0972-1363.145022

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   Abstract 

Keratocystic odontogenic tumor (KCOT) is a benign intraosseous neoplasm of the jaw with a high rate of recurrence. The lesion commonly occurs in the mandibular molar-ramus area and is rarely seen in the maxilla. Its occurrence in the maxillary sinus along with a displaced third molar is very rare. This article reports a case of KCOT in a 15-year-old boy with a displaced third molar involving the right maxillary antrum.

Keywords: Displaced maxillary third molar, KCOT, maxillary antrum


How to cite this article:
Mengji AK, Yaga US, Besta R, Doshetty RS. Keratocystic odontogenic tumor involving the maxillary antrum with displacement of the third molar: A rare case report . J Indian Acad Oral Med Radiol 2014;26:335-7

How to cite this URL:
Mengji AK, Yaga US, Besta R, Doshetty RS. Keratocystic odontogenic tumor involving the maxillary antrum with displacement of the third molar: A rare case report . J Indian Acad Oral Med Radiol [serial online] 2014 [cited 2022 May 22];26:335-7. Available from: https://www.jiaomr.in/text.asp?2014/26/3/335/145022


   Introduction Top


Philipsen, in the year 1956, first described the term 'odontogenic keratocyst'. However, this entity was renamed as 'Keratocystic odontogenic tumor' by the World Health Organization (WHO) in the year 2005. These tumors are developmental, noninflammatory, and odontogenic in origin, arising from the cell rests of the dental lamina. [1],[2],[3] About 70-80% of the tumors occur in the lower jaw, out of which, about 90% occur posterior to the canines and 50% in the ascending ramus of the mandible. They occur very rarely in the upper jaw. [4],[5],[6]

The tumors are often confused with ordinary cysts, because of the nonspecific clinical and radiographic features. This in turn, leads to a misdiagnosis and improper treatment, resulting in unnecessary recurrences. Successful treatment depends on the precise diagnosis, adequate surgical procedure, and follow-up. [7] The objective of this article is to enlighten a rare case of KCOT located in the right maxillary sinus, in a 15-year-old boy, which had also displaced the third molar.


   Case Report Top


A 15-year-old boy visited the Department of Oral Medicine and Radiology, with a chief complaint of pus discharge from the right posterior region of the upper jaw since three years. At the age of 12 years, the patient noticed pus discharge from the same region, which was sudden in onset and intermittent in nature. The episode of events continued during these years, which got aggravated one month before the patient reported to us. On extraoral examination, there were no marked findings except for a mild diffuse swelling on the right side of the face involving the right maxillary sinus region, which was tender to palpation. The lymph nodes were not palpable on both the sides. On intraoral examination a diffuse bony swelling was seen on the buccal aspect of the alveolus, obliterating the vestibule, and extending from the mesial border of 16 posteriorly beyond the zygomatic buttress area [Figure 1]a. On palpation, mild tenderness was present. In relation to the upper right second molar region on the palatal mucosa, 1 cm away from the attached gingiva, there was a small sinus opening, about 0.5 to 1 mm diameter, without any visible discharge [Figure 1]b. Exertion of mild pressure in this region also did not elicit any discharge; however, there was mild tenderness. Vitality tests were performed on teeth 14, 15, 16, and 17, which showed that the teeth were vital. Tooth 18 was missing. Considering the clinical history and findings, a provisional diagnosis of infected dentigerous cyst involving the right upper third molar was made. Maxillary sinusitis (as the lesion was close to the sinus area) and odontogenic keratocyst were included under the differential diagnoses.
Figure 1: Image showing the (a) intraoral view and (b) the region of the palatal sinus opening distal to the right maxillary second molar

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The panaromic radiograph (OPG) revealed an irregular osteolytic lesion extending anteroposteriorly from the distal root of the upper right second molar to the tuberosity, and superoinferiorly, from the sinus region to the alveolar crest region of 16 and 17. The upper right third molar was displaced superiorly to the roof of the maxillary sinus [Figure 2]a. The paranasal sinus (PNS) view of the skull revealed haziness in the right maxillary antrum region, with a displaced third molar in the superomedial position of the maxillary antrum [Figure 2]b. Computed tomography (CT) revealed a hyperdense mass in the maxillary antrum, with displacement of maxillary third molar toward the roof of the sinus, displacement of the nasal septum toward the left side, and perforations in the buccal and palatal cortical plates in relation to the right maxillary second molar [Figure 2]c.
Figure 2: Radiographic findings in the (a) OPG showing an irregular osteolytic lesion (b) PNS view of skull showing haziness in the right maxillary antrum, and (c) CT showing a hyperdense mass in the right maxillary antrum

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Aspiration yielded pus with blood [Figure 3]a. Enucleation of the lesion along with extraction of the displaced third molar was performed under general anesthesia [Figure 3]b. Histopathological examination showed six-to-ten cell thick epithelial corrugation, a parakeratinized stratified epithelium, a palisaded hyperchromatic basal layer, with an archading pattern, nests of odontogenic epithelial rests, and daughter cysts [Figure 3]c and d. The histopathological findings were suggestive of KCOT. A follow-up after six months with radiographic evaluation including OPG, PNS view of the skull, and CT revealed no evidence of recurrence.
Figure 3: Image showing the (a) aspirated fl uid (b) enucleated mass with the extracted upper third molar (c) photomicrograph with six-toten cell thick epithelial corrugation, parakeratinized stratifi ed epithelium, with a palisaded hyperchromatic basal layer, archading pattern, nests of odontogenic epithelial rests, daughter cysts, and (d) keratin pearls

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   Discussion Top


The odontogenic keratocyst was first described in 1876. It was named by Phillipsen in the year 1956. It is one of the most aggressive odontogenic tumors of the oral cavity and has a rapid growth. It has a tendency to invade the adjacent tissues, including the bone. It has a high recurrence rate and is associated with the basal cell nevus syndrome. [8]

The keratocystic odontogenic tumor is more common in males than females and occurs over a wide age range. However, it is typically diagnosed during the second to fourth decade. [9] In our case, the patient was in his second decade. KCOTs mostly occur in the body of the mandible, most commonly in the molar region and vertical ramus. [2] However, in the present case, there was maxillary antrum involvement, with displacement of the right maxillary third molar. The case reported here shows a rare site of occurrence in a 15-year-old boy. Literature suggests that less than 1% of the KCOT cases occur in the maxilla with maxillary antrum involvement. In the maxilla, the canine region is the most common location for KCOTs.

The clinical features include pain, soft-tissue swelling, expansion of bone, drainage, and various neurological manifestations, such as, paresthesia of the lip or teeth in case of lower jaw involvement. The maxillary KCOT tends to be secondarily infected with greater frequency than the mandibular ones, due to its vicinity to the maxillary sinus. [10] In this case too, the lesion was secondarily infected, with the presence of a sinus opening and pus discharge.

Radiographically most KCOTs are unilocular, presenting a well-defined peripheral rim, with scalloped borders. Multilocular radiolucent area generally represents a central cavity having satellite cysts. [11] KCOTs may be mistakenly diagnosed as inflammatory lesions of endodontic origin or lateral periodontal cysts. In the present case, the radiographic evaluation revealed an aggressive osteolytic lesion, which not only displaced the third molar, but also caused perforations in the buccal and palatal cortical plates in the region of 17. Histologically, these cysts show a stratified squamous epithelium that produces orthokeratin (10%), parakeratin (83%), and or both types (7%). The case presented here was of the parakeratin type.

Numerous surgical modalities have been suggested for the treatment of KCOTs, including, enucleation with primary closure, enucleation with open packing, marsupilization, enucleation with use of Carnoy's solution or cryotherapy, with a marginal or radical section. Although KCOTs are stated to be the most aggressive and recurrent form of tumors, there are cases where KCOTs have been treated by enucleation. In the present case also it has been noted that there has been complete healing following the enucleation procedure. Nevertheless, a long-term follow-up is required to ascertain non-recurrence of the tumor. [12]


   Conclusion Top


A case of KCOT occurring in a rare site is reported here. With the nonspecific, clinical, and radiological findings (in the conventional radiographs), the lesion presented here could easily be misdiagnosed as an ordinary cyst or an inflammatory process. Advanced imaging techniques such as cone-beam CT should be advised in such cases for accurate diagnosis and appropriate treatment. In addition to histopathological confirmation, a long-term follow-up is a must, considering the high recurrence rate of KCOT.

 
   References Top

1.Shear M, Speight P. Odontogenic keratocyst. In: Shear M, Speight P, editors. Cysts of the Oral and Maxillofacial Regions. 4 th ed. Oxford: Blackwell Munksgaard; 2007. p. 6-58.  Back to cited text no. 1
    
2.Madras J, Lapointe H. Keratocystic odontogenic tumor: Reclassification of the odontogenic keratocyst from cyst to tumor. J Can Dent Assoc 2008;74:165-165h.  Back to cited text no. 2
    
3.O'Neill R, Al-Hezaimi K. Identification of an odontogenic keratocyst and treatment with guided tissue regeneration: Case report. J Can Dent Assoc 2011;77:b6.  Back to cited text no. 3
    
4.Pazdera J, Kolar Z, Zboril V, Tvrdy P, Pink R. Odontogenic keratocysts/keratocystic odontogenic tumors: Biological characteristics, clinical manifestation and treatment. Biomed Pap Med Fac Univ Palacky Olomouc Czech Repub 2014;158:170-4.  Back to cited text no. 4
    
5.White SC, Pharoah MJ. Cysts of the jaws. In: White SC, editor. Oral Radiology: Principles and Interpretation. 5 th ed. St. Louis: Mosby Elsevier; 2009. p. 395-7.  Back to cited text no. 5
    
6.Jafaripozve S, Allameh M, Khorasgani MA, Jafaripozve N. Keratocyst odontogenic tumor in the anterior of the maxilla: A case report and literature review. J Oral Maxillofac Radiol 2013;1:90-2.  Back to cited text no. 6
  Medknow Journal  
7.Silva GC, Silva EC, Gomez RS, Vieira TC. Odontogenic keratocyst in the maxillary sinus: Report of two cases. Oral Oncol EXTRA 2006;42:231-4.  Back to cited text no. 7
    
8.Oda D, Rivera V, Ghanee N, Kenny EA, Dawson KH. Odontogenic keratocyst: The northwestern USA experience. J Contemp Dent Pract 2000;1:60-74.  Back to cited text no. 8
    
9.Ortakoðlu K, Süer BT, Þençimen M. A large odontogenic keratocyst containing a third molar tooth in the maxillary antrum. Turk J Med Sci 2005;35:341-6.  Back to cited text no. 9
    
10.Rajendran R, Sivapathasundaram S. Shafer's Textbook of Oral Pathology. 6 th ed. New Delhi: Elsevier Publication; 2009. p. 258-60.  Back to cited text no. 10
    
11.Ali M, Baughman RA. Maxillary odontogenic keratocyst: A common and serious clinical misdiagnosis. J Am Dent Assoc 2003;134:877-83.  Back to cited text no. 11
    
12.Pazdera J, Kolar Z, Zboril V, Tvrdy P, Pink R. Odontogenic keratocysts/keratocystic odontogenic tumors: Biological characteristics, clinical manifestation and treatment. Biomed Pap Med Fac Univ Palacky Olomouc Czech Repub 2014;158:170-4.  Back to cited text no. 12
    


    Figures

  [Figure 1], [Figure 2], [Figure 3]


This article has been cited by
1 Large Follicular Odontogenic Keratocyst affecting Maxillary Sinus mimicking Dentigerous Cyst in an 8-year-old Boy: A Case Report and Review
Madhusudhan R Madhireddy, A Jacob Prakash, Vijayalakshmi Mahanthi, K Venkata Chalapathi
International Journal of Clinical Pediatric Dentistry. 2018; 11(4): 349
[Pubmed] | [DOI]



 

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