Maxillary sinus harbours many pathological lesions. Many of those presents as a sinonasal mass and are rarely symptomatic. These masses are usually an antral polyp, mucoceles or mucous retention cysts. Odontogenic keratocyst, a benign odontogenic lesion presenting within the maxillary sinus is a rare entity. We present a case of odontogenic keratocyst of the maxillary sinus in a 35 years old female

Geethika, V

Kumar, PK

Ramaswamy, P

Sreenivasulu, P

Sudhakar, S

Cognitive Sciences ePrint Archive

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Case Report: 
A Rare Presentation of Odontogenic Keratocyst Mimicking an Antral Polyp. 
Authors 
S Sudhakar, Reader, 
V Geethika,Post graduate Student, 
P Ramaswamy, Professor & HOD, 
B Praveen Kumar, Reader, 
P Sreenivasulu, Senior Lecturer 
Department of Oral medicine & Radiology, St.Joseph dental college & Hospital, Eluru, Andhrapradesh, India. 
 
Address for Correspondence 
Dr. Sudhakar S, 
Reader, Department of Oral medicine & Radiology, 
St.Joseph dental college & Hospital, 
Eluru, Andhrapradesh, India.  
E-mail: drsudhakaroralmed@yahoo.co.in 
 
Citation 
Sudhakar S, Geethika V, Ramaswamy P, Kumar PK, Sreenivasulu P. A Rare Presentation of Odontogenic Keratocyst Mimicking an 
Antral Polyp. Online J Health Allied Scs. 2013;12(4):10. Available at URL: http://www.ojhas.org/issue48/2013-4-10.html 
 
Open Access Archives 
http://cogprints.org/view/subjects/OJHAS.html 
http://openmed.nic.in/view/subjects/ojhas.html 
Submitted: Nov 10, 2013; Accepted: Jan 4, 2014; Published: Feb 20, 2014 
 
Abstract: Maxillary sinus harbours many pathological 
lesions. Many of those presents as a sinonasal mass and are 
rarely symptomatic. These masses are usually an antral 
polyp, mucoceles or mucous retention cysts. Odontogenic 
keratocyst, a benign odontogenic lesion presenting within the 
maxillary sinus is a rare entity. We present a case of 
odontogenic keratocyst of the maxillary sinus in a 35 years 
old female. 
Key Words: Keratocyst; Maxillary sinus 
Case Report: 
A 35 years old female patient reported with a complaint of 
discomfort on the upper right cheek region since 15 days. 
Patient history suggested that she had similar complaints for 
the past three months and had subsided upon taking some 
over the counter medications. The patient claims the present 
complaint is since 15 days and had not subsided upon taking 
the same medications. Although the discomfort was not 
debilitating nor it is causing any functional disability she 
reported to us in search of a permanent relief. Her medical, 
surgical and family histories were non-contributory. The past 
dental history revealed history of extraction of upper right 
second premolar in a private dental clinic a year ago, 
following dental caries and periapical infection. The post-
operative period was uneventful. 
On extraoral examination mild tenderness over the right 
maxillary sinus region was elicited. Nares and nasal septum 
appeared normal. Intraoral examination showed missing right 
second premolar and a scar/ indentation noticed over the 
premolars vestibular space with mild tenderness on palpation. 
There was no evidence of vestibular obliteration, expansion 
or pus discharge (Fig 1). 
 
Fig 1: Intraoral photograph showing the normal 
vestibular spaces 
 
Fig 2: OPG showing the healing defect over the right 
premolar region 
 
This work is licensed under a 
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Volume 12, Issue 4; Oct-Dec 2013 
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Fig 3: PNS view showing a radiopaque spherical mass 
involving the right maxillary sinus 
 
Fig 4: Computed tomography picture showing a well 
defined spherical area involving the right maxillary sinus 
 
Fig 5: Intra-operative photograph 
Patient was then subjected to routine orthopantomograph 
(OPG) and paranasal sinus (PNS) view. The OPG showed 
missing right second premolar with an arc shaped bone loss 
over the right first premolar extending from the level of 
alveolar crest towards the junction of cervical and middle 
third of the root (Fig 2). The PNS view showed a well-
defined pyramidal shaped radiopaque mass ranging 1.5x1 cm 
in size located at the core of the right maxillary antrum (Fig 
3). The computed tomography picture showed presence of a 
well defined homogenous spherical mass ranging 2x2 cm 
within the confines of the right maxillary sinus. The 
surrounding area appeared normal with no evidence of bony 
destruction (Fig 4). The features were suggestive of a 
polypoid mucosal mass within the right antrum. 
 
Fig 6: Photomicrograph of the excised specimen 
 
Fig 7: Post-op follow up paranasal sinus view 
Considering the symptomatic nature of the mass, the panel of 
experts comprising oral physician, oral surgeon and 
otorhinolaryngologist decided to surgically explore the 
lesion. Under general anesthesia, the mass was approached 
through maxillary antrostomy and completely enucleated 
(Fig 5). The post-operative period was uneventful. 
Histopathological analysis of the excised mass showed 
presence of pseudostratified ciliated columnar epithelium 
with subepithelial glands, numerous capillaries, inflammatory 
infiltrates and bony trabeculae. Focal areas showed presence 
of stratified parakeratinized corrugated epithelium of 5-6 
layers thickness with palisading of basal tall columnar cells. 
The connective tissue shows mild chronic inflammatory cells 
(Fig 6). These features were suggestive of odontogenic 
keratocyst- parakeratinized variant. 
Considering the above diagnosis the patient was recalled and 
cautioned about the recurrent nature of the lesion. Till date 
there is report of recurrence and the patient is kept under 
every six months follow-up (Fig 7). 
Discussion: 
The diseases involving the sino-nasal regions are manifold 
and it includes developmental, infectious, traumatic and even 
malignancies. Interestingly the clinical presentations too 
3 
 
range from completely asymptomatic to symptoms like pain, 
swelling, obstructions, discharge and altered smell etc., The 
commonest sino-nasal disorders are sinusitis, allergic rhinitis 
and antrochoanal polyps. The sinusitis and rhinitis are 
inflammatory lesions of their respective regions whereas the 
antrachonal polyp is considered to be its sequel. 
The common differential diagnosis of ACPs should include 
juvenile angiofibroma, nasal glioma, meningoencephalocele, 
inverted papilloma, mucocele, mucus retention cyst, 
Tornwalt’s cyst, grossly enlarged adenoids, lymphoma and 
nasopharyngeal malignancies.(1-6) Presence of odontogenic 
keratocyst (OKC), a lesion arising from the odontogenic 
apparatus, a completely non-native entity in the sinonasal 
region is a rarity. Until now, only two cases of OKC present 
completely within the maxillary sinus has been reported in 
the literature.(7) 
OKC, first described by Philipsen in 1956 and later designed 
by the World Health Organization as keratocystic 
odontogenic tumor (KCOT), is a benign uni or multicystic 
intraosseous tumor of odontogenic origin, with a 
characteristic lining of parakeratinized squamous 
epithelium.(8)  
The origin of OKC is suggested to be from the dental lamina 
remains.(9) However, some author’s support that it should be 
considered a benign cystic neoplasm related to the mutation 
of tumor suppressor gene, PTCH and more recently, 
intracystic fluid pressure was found to be involved in OKC 
growth.(10,11) 
OKC comprises approximately 11% of all cysts of the jaws. 
The OKC has a predilection for men and are common in 
second and third decades of life. They occur most commonly 
in the mandible, especially in the posterior body and ramus 
regions.(12,13) They almost always occur within the bone, 
although a small number of cases of peripheral variants have 
also been reported.(8) 
Reports suggests that less than 1% of all cases of OKC occur 
in the maxilla with the involvement of the maxillary 
sinus.(13,14) In the above said cases, the OKC’s are found to 
be primarily developing from the maxillary bone then 
extends to involve the maxillary sinus. However, rare 
presentations of OKC completely restricted within the 
maxillary sinus without the involvement of the alveolar bone 
or in association with an unerupted teeth have also been 
reported.(7) 
The case reported here also showed OKC present within the 
sinus but more interestingly without the association of any 
unerupted/ impacted tooth. Usually patients with OKC 
presents with swelling, pain and discharge. Distinctive 
clinical features include a potential for expansion, local 
destruction and a tendency for multiplicity, our patient did 
not have any such presentation.(15) 
Radiographically, intraosseous odontogenic keratocyst 
present as a multilocular or unilocular radiolucency with 
well-developed sclerotic borders. As OKC appearance within 
the maxillary sinus is rare, its radiographic image in such 
situation may be misinterpreted, as seen with the present 
case. Although computed tomography can provide 
information on the extent of these lesions, due to relative lack 
of specific characteristics it may not contribute to diagnosis 
and preoperative preparation.(9) Contrast enhanced MR 
imaging can provide the essential macroscopic detail, 
including focal wall enhancement and iso-intense 
intraluminal soft tissue mass, which correlates with the 
histological findings of focal inflammatory ulceration of the 
cyst lining, cytokeratin and cell debris.(16) In many instances 
only CT is advised, only rarely both CT and MRI are advised 
that too when there is a gross destruction of bone, since the 
distinction between them is more of an intellectual interest 
rather than clinical significance. Unfortunately in the present 
case too only CT was advised. Histologically, OKCs are 
formed with a stratified squamous epithelium that produces 
orthokeratin, parakeratin or both. The epithelial lining 
appears corrugated, with a wellpolarized hyperchromatic 
basal layer and the cells remain basaloid almost to the 
surface. No rete ridges are present; therefore, the epithelium 
often sloughs from the connective tissue. 
The epithelium is thin and mitotic activity is frequent; 
therefore, OKCs grow in a neoplastic fashion and not in 
response to internal pressure. The lumen is frequently filled 
with a foul smelling cheese-like material that is not pus but 
rather collected degenerating keratin.(8) Various treatment 
alternatives based on surgical approaches have been 
suggested, such as marsupialization, enucleation, enucleation 
with Carnoy’s solution, enucleation with cryotherapy, 
curettage and resection. Simple enucleation was associated to 
a higher recurrence rate, while resection and enucleation with 
bone curettage presented lower rates. Special attention should 
be given to the dentate area if the enucleation is chosen as 
treatment, due to higher rates of recurrence found in OKC 
associated with teeth.(7)  In the present case enucleation was 
done based on the initial judgement of the lesion, however 
the patient is under regular follow up with no evidence of any 
recurrence till date. 
Conclusion 
Odontogenic keratocyst is an uncommon entity in the 
maxillary sinus region. If involved it usually extends from 
the underlying maxillary bone then secondarily into the 
sinonasal region. The case presented here is a rarity as neither 
there is a gross destruction of the underlying bone nor any 
associated impacted tooth and the lesion is completed within 
the sinus. The histopathological analysis suggested the 
presence of squamous epithelium and no evidence any 
sinonasal comorbidity. 
References: 
1. Towbin R, Dunbar JS, Boven K. Antrachoanal 
polyps. Am J Roentgenol. 1979;132(1):27-31. 
2. Basak S, Karaman CZ, Akdilli A, Metin KK. Surgical 
approaches to antrochoanal polyps in children. Int J 
Pediatr Otorhinolaryngol. 1998;46(3):197-205. 
3. Lee TJ, Huang SF. Endoscopic sinus surgery for 
antrochoanal polyps in children. Otolaryngol Head 
Neck Surg. 2006;135(5): 688-92. 
4. Ozdek A, Samim E, Bayiz U, Meral I, Safak MA, Oguz 
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5. Yuca K, Bayram I, Kiroglu AF, Etlik O, Cankaya H, 
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13. Brannon RB. The odontogenic keratocyst: a 
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A Rare Presentation of Odontogenic Keratocyst Mimicking an Antral Polyp

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