Mrudul M S, Sunil S, Baby Tibin K, Babu Sharlene Sara

Department of Oral and Maxillofacial Pathology, Pushpagiri College of Dental Science, Tiruvalla, Kerala

Running title – Odontogenic Keratocyst in maxillary sinus

Received: 5-09-2023

Revised: 7-09-2023

Accepted: 12-09-2023

Address for correspondence: Dr.Tibin K. Baby, Reader, Department of Oral & Maxillofacial Oral Pathology, Pushpagiri college of Dental Science, Tiruvalla, Kerala

Email: tibinkbaby@gmail.com

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How to cite this article: Mrudul M S, Sunil S, Baby Tibin K, Babu Sharlene Sara. Odontogenic Keratocyst in the Maxillary Sinus:  A Rare Case Report. J Oral Biomed Sci 2023; 2(3):113-118

ABSTRACT

The odontogenic keratocyst (OKC), a developmental,   potentially aggressive odontogenic cyst having the highest reccurence rate and pathognomonic histopathological features. Most cased reported are located in posterior mandibular region. The occurrence of OKC in the posterior maxillary region is not common and its incidence in the maxillary sinus very rare. We describe a case of OKC in the right maxillary sinus region that had no clinical signs but was diagnosed early and successfully treated immediately preventing complications.

Keywords:  odontogenic keratocyst, maxillary sinus, picket fence, Carnoy’s solution

INTRODUCTION

Odontogenic keratocyst (OKC) is a cystic lesion of odontogenic origin with aggressive and infiltrative characteristics. It accounts for 10% of odontogenic cysts and 80% of cases occurred in the mandibular posterior region. However, it is also present, particularly in the anterior, canine region of the maxilla. Less than 1% of OKC cases occured in the maxillary sinus region.1,2,3,4,5 Dental lamina remnants give rise to OKCs, which can develop as a sole lesion or as a cluster of cysts and are associated with disorders like Gorlin-Goltz.6   OKC was categorised as a keratocystic odontogenic tumour (KCOT) by the World Health Organization (WHO) in 2005. The WHO reclassified KCOT back to OKC in 2017.8  Being  nonspecific in  clinical and radiographic characteristics, it may be mistaken for a regular cyst, resulting in a missed diagnosis and inadequate treatment,  causing unwanted recurrences. Prompt diagnosis, a suitable surgical method, and follow-up are essential for a successful prognosis.9,10 We present a rare case report  of OKC  in the maxillary sinus highlighting the importance of early diagnosis and prompt treatment.

CASE REPORT

A 19-year-old male came for orthodontic treatment with no clinical signs and symptoms of any pathologic condition. Hard-tissue examination showed missing 18.  Orthopantomogram revealed a well-defined, radiolucent lesion related to an impacted third molar displaced to the right maxillary sinus extending from distal side of 14 to mesial side of 16.  CT scan revealed the impacted 18 and OKC in the floor extending to the maxillary sinus roof. Under general anesthesia, lesion was enucleated along with the impacted tooth. Excisional biopsy showed a cystic capsule with parakeratinized stratified squamous epithelium of 6–10 layers of thickness and surface corrugation. Nuclear palisading and hyperchromatism were present in the basal cells. The epithelial connective tissue junction was flat and had localised regions of separation. The cystic wall appeared fibrous with dense infiltration of inflammatory cells. The histopathologic features confirmed the diagnosis of OKC.

Figure 1: Orthopantamogram showing impacted 18 with a radiolucent lesion extending from 14 to 17 region and roof of right  maxillary sinus.

Figure 2: showing Computed Tomography scan showing radiolucency and impacted 18 in the right maxillary sinus

Figure 3 : showing excised cystic lesion with impacted 18

Figure 4: Photomicrograph showing cystic capsule lined by uniform thickness of epithelium [ 4X].

Figure 5: Photomicrograph showing cystic capsule lined by uniform thickness of 6-10 layered epithelium with palisaded basal layer,  [10X].

Figure 6: Photomicrograph showing cystic capsule lined by uniform thickness of 6-10 layered epithelium with palisaded basal layer,  [40X].

DISCUSSION

Odontogenic keratocyst (OKC), first described by Philipsen[1956],was also called primordial cyst,  is an aggressive developmental odontogenic cyst [ 4-12 %], derived from rests of dental lamina.1-3

OKC often results in expansion of the jaw associated with a unilocular or more commonly multilocular radiolucency. Posterior mandicle is the most common site. Lesions are usually solitary.3-5

It can occur at any age, but peak in the second and third decades of life with slight  male predilection. Posterior mandible is more commonly affected. The common clinical features include pain, soft tissue swelling, bone expansion,nasal obstruction, tooth mobility, drainage, paresthesia, OKC have an anteroposterior growth pattern.1-5,12-15

In the radiograph, OKC appears commonly as unilocular radiolucency with well-defined rim or scalloped border or may be multilocular radiolucent lesion.18

Genetic study reveals Two-hit mechanism resulting in bi-allelic loss of PTCH (“patched”) tumor suppressor on 9q22.3-q31 causing dysregulation of p53 and cyclin D1 oncoproteins, rarely associated with Nevoid Basal Cell Carcinoma Syndrome / Gorlin Syndrome.32

Its aggressiveness and high recurrence rate is characteristic. OKC showed higher levels of p53. PTCH2 and SUFU, p16, MCC, TSLC1, LTAS2, and FHIT are the other genes expressed. Three histologic variants are parakeratinized, orthokeratinized and a combination of the two33

The histopathologic findings include a cystic capsule which is loose to moderately collagenous with 6-10 cell layer lining para/ortho keratinized epithelium having corrugated surface. There is a prominent palisaded polarized basal layer with a tombstone or picket fence appearance. The capsule may have islands of epithelium or small/ daughter/ satellite cysts. The lumen shows cheesy material of keratin or sometimes contain straw coloured fluid.2

Usually multiple OKC lesions may be associated with nevoid basal cell syndrome (Gorlin–Goltz syndrome).16 The continued growth of an odontogenic keratocyst can encroach the sinus space and displace its borders. The displacement of tooth buds caused by cyst expansion can lead to “ectopic” eruption of a tooth.15

OKC can resemble dentigerous cyst or ameloblastoma both clinically and radiographically. Hence proper histopathologic evaluation is mandatory for proper diagnosis and treatment.22

OKC subtypes include parakeratinized,orthokeratotic or a combination of the two. The most aggressive and frequent (80%) subtype is para keratotic with high chance for recurrence.21

Treatment option of resection had a recurrence rate of 0 %, and enucleation with adjunctive therapies with Carnoy’s solution and marsupialization  had recurrence rate 1–8.7 %, while enucleation alone had recurrences of 17–56 %.25-31

Several factors should be taken into account in order to achieve successful treatment. Factors influencing treatment include the size, location, uni- or multilocularity, soft tissue involvement, cortical perforation, patient characteristics such as age, and adequate surgical procedure and follow-up.12,13,26

CONCLUSION

It is rare to have OKC in the maxillary sinus, and when it does, it typically does not exhibit the same clinical and radiographic characteristics as its other counterparts within the jaw bone. Its potential for aggressive behaviour and recurrence of OKC sets it apart from other jaw cysts. Because of the aggressive nature and high reccurence nature of odontogenic keratocyst, early diagnosis and appropriate treatment is essential to prevent complications and recurrence.

Acknowledgement: Dr. Arun B Nair, ENT Surgeon, Gitanjali Hospitals, Trivandrum.

Conflict of interest: None

Source of support: Nil

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