Open Access Case

Report DOI: 10.7759/cureus.37357

An Unusual Case of Maxillary Sinus Odontogenic

Keratocyst: An Insightful Report With Review of
Review began 04/03/2023
the Literature
Review ended 04/06/2023
Published 04/10/2023 Shruti Singh 1 , Priti Shukla 2 , Ravinder S. Bedi 3 , Shruti Gupta 4 , Shivesh Acharya 5

© Copyright 2023
Singh et al. This is an open access article 1. Oral and Maxillofacial Pathology, All India Institute of Medical Sciences, Raebareli, IND 2. Orthodontics and
distributed under the terms of the Creative Dentofacial Orthopaedics, All India Institute of Medical Sciences, Raebareli, IND 3. Oral and Maxillofacial Surgery, All
Commons Attribution License CC-BY 4.0., India Institute of Medical Sciences, Raebareli, IND 4. Pathology, All India Institute of Medical Sciences, Raebareli, IND
which permits unrestricted use, distribution, 5. Pediatric Dentistry, All India Institute of Medical Sciences, Raebareli, IND
and reproduction in any medium, provided
the original author and source are credited.
Corresponding author: Priti Shukla, drpritishukla22@gmail.com

Abstract
Maxillary sinus odontogenic keratocyst (OKC) is very rare and occupies less than 1% of the total
OKC cases reported in the literature. OKCs have characteristic features that are unique compared to other
cysts of the maxillofacial region. Considering their peculiar behaviour, varied origin, debated development,
discourse treatment modalities, and high recurrence rate, OKCs have been a subject of interest for various
oral surgeons and pathologists globally. This case report presents an unusual case of invasive maxillary
sinus OKC into the orbital floor, pterygoid plates, and hard palate in a 30-year-old female. The case
report confers that cystic maxillary sinus lesions should always be treated very extensively irrespective of the
nature of the lesion as the site makes it highly susceptible to secondary infection and recurrence. The case
also establishes a set of imaging modalities and specific treatment approaches to be followed for maxillary
sinus OKC based on the literature of all the previous cases reported.

Categories: Pathology, Dentistry, Oral Medicine

Keywords: contrast-enhanced ct, pterygoid plate, carnoy’s solution, odontogenic keratocyst, maxillary sinus

Introduction
Odontogenic keratocyst (OKC) is a distinctive form of developmental odontogenic cyst comprising 12% of
the entire jaw cysts [1]. Considering their peculiar behaviour, varied origin, debated development, discourse
treatment modalities, and high recurrence rate, OKCs have been a subject of interest for various oral
surgeons and pathologists globally [2].

The most common site as per the occurrence rate of OKC is the mandible (73%) compared with 27% in the
maxilla [3]. OKCs of the maxillary sinus are even rarer with less than 1% of cases reported in the literature
[4].

This case report is of an unusual presentation of an aggressive left maxillary sinus OKC in a 30-year-old
female. Lesions involving the sinus always pose a diagnostic challenge as the margins are difficult to identify
and sinus pathology of odontogenic origin holds high chances of secondary infection [5]. A brief review of
all the reported maxillary sinus OKCs of the past 20 years has also been compiled with this case for
understanding their behaviour patterns in terms of clinical, radiological, and treatment outcomes
considering its rare occurrence in this site.

Case Presentation
A 30-year-old female patient reported to the department of dentistry with the chief complaint of swelling on
the left upper back region of the jaw for three years with mild heaviness for a month on the same side of the
face. The patient gave a history of gradual increase in the size of the swelling to the present size with no
association of pain or any other symptom. No significant personal, medical, or family history was reported.
Slight facial asymmetry was present extraorally on the affected side (Figure 1).

How to cite this article

Singh S, Shukla P, Bedi R S, et al. (April 10, 2023) An Unusual Case of Maxillary Sinus Odontogenic Keratocyst: An Insightful Report With Review
of the Literature. Cureus 15(4): e37357. DOI 10.7759/cureus.37357
 FIGURE 1: Extraoral clinical image
Extraoral clinical image showing no visible gross facial asymmetry.

Intraoral examination revealed diffuse swelling on the buccal aspect of the maxilla completely obliterating
the vestibule region of 23-27 extending palatally involving the hard palate (Figure 2).

FIGURE 2: Diffuse swelling on the buccal aspect of the maxilla

Diffuse swelling on the buccal aspect of the maxilla completely obliterating the vestibule region of 23-27.

On palpation, the swelling was non-tender and firm in consistency. Hard tissue examination revealed no

2023 Singh et al. Cureus 15(4): e37357. DOI 10.7759/cureus.37357 2 of 11

 clinical decay or mobility in the tooth in relation to the swelling, nor was there any history of discharge. The
vitality of the teeth was assessed, and all the teeth were vital. Chronic generalized periodontitis with the
presence of dental fluorosis was seen (Figure 2). Considering the site and extent of the swelling on intraoral
examination without any oral symptoms, a clinical differential diagnosis of maxillary odontogenic sinusitis,
periodontal cyst, and deep fungal infection of the maxillary sinus was considered. Hence, an
orthopantomogram (OPG) (Figure 3) and a paranasal sinus view followed by contrast-enhanced computed
tomography (CECT) were advised. The paranasal sinus view showed complete obliteration of the left
maxillary sinus (Figure 4).

FIGURE 3: Pre-treatment orthopantomogram showing the expansion of

the sinus lining

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 FIGURE 4: Paranasal sinus view showed complete obliteration of left
maxillary sinus

CECT findings revealed an expansile lytic lesion in the left upper alveolus with cortical destruction.
Hypodense non-enhancing soft tissue component was reported involving the roots of the teeth of the upper
left posterior quadrant. The medial and lateral wall of the maxillary sinus was involved (Figure 5).

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 FIGURE 5: (A) Coronal and (B) axial CT: the expansile lytic lesion in the
left alveolus with hypodense non-enhancing soft tissue component

The lesion extended superiorly up to the orbit and medially involved the complete maxillary left sinus,
inferiorly up to the hard palate and laterally up to the masseteric space (Figure 5A).

CT report concluded it to be a benign cystic lesion. Fine needle aspiration cytology (FNAC) was done, and
aspirate yielded cream-coloured fluid and cytology revealed the presence of necrosis with cholesterol
crystals.

The surgical enucleation of the lesion was planned under local anaesthesia and antibiotic coverage with an
intra-oral approach (Figure 6).

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 FIGURE 6: Surgical exploration through intra-oral approach: thinning
and expansion of cortical plates

During surgical exploration, an abundance of white-cheesy content was collected. The lesion was highly
aggressive with invasion into the pterygoid plates and orbital floor. The fragile cyst lining was enucleated,
and curettage was done. Considering the invasive nature of the lesion, extended chemical cauterization was
done using freshly prepared Carnoy’s solution. The cyst cavity was inspected to ensure complete excision,
and an iodoform-medicated gauze pack was kept inside the cavity with one end out. This gauze pack was
removed after three days.

The enucleated specimen was sent for histopathological examination. Gross examination showed a thin cyst
wall, which on microscopy revealed a cystic lining with parakeratinized stratified squamous epithelium with
palisaded cuboidal basal cells. The cystic wall was fibrous with focal areas of dense chronic inflammatory
infiltrate predominantly plasma cells and lymphocytes (Figure 7). Therefore, the histopathological diagnosis
of OKC of the maxillary sinus was made.

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 FIGURE 7: Fibro-dense cyst wall lined with para keratinization stratified
squamous lining with palisaded cuboidal basal cells. The cystic wall is
fibrous with focal areas of moderately dense chronic inflammatory cells

The patient was followed up after four months and had no fresh complaints in accordance with the site of
the lesion operated, and intraorally the site seemed to have restored its normal anatomy (Figure 8).

FIGURE 8: Post-operative follow-up image after four months post-

surgery

The patient was kept in periodic follow-up, and OPG was repeated six months post-surgery, which showed
no recurrence (Figure 9). CECT is to be repeated at the one-year post-surgery visit.

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 FIGURE 9: Post-operative orthopantomogram at six months follow-up
showing no recurrence

Discussion
OKCs are the third most common developmental jaw cysts. The term was broadly used for all keratin-
forming cysts during the 1950s. It was in 1956 that Phillipsen described OKC for epithelial developmental
cysts [6]. OKCs show an occurrence of 65% to 83% in the mandible but their location in the maxillary site is
conflicted [2,3]. OKCs that occur in regions other than the mandibular angle and especially those in the
maxilla seem to be more related to systemic syndromes [3]. A literature search for case reports was
conducted in PubMed, ClinicalKey, and Google Scholar databases using the keywords "maxillary sinus" and
"invasive odontogenic keratocyst" and the available data for the last 20 years have been tabulated (Table 1).

Panoramic
Computed tomography
Year Age/gender Clinical features radiographic Treatment and recurrence
findings
findings

Case 1 presented
Case 1: Lateral and
with symptoms of
post-anterior Case 2: Ectopic second
headache from a Both were enucleated and
Case 1: 17 radiographs showed a right upper molar
week with no curettage was done. Case 1:
Silva et al. years/male; discrete opaque mass involved by radiopacity
swelling. Case 2 Follow-up of eight years with no
(2006) [7] Case 2: 14 with an image of the filling in the upper
presented with recurrence. Case 2: Follow-up of
years/boy third molar in the left posterior portion of the
intra-oral swelling five years with no recurrence.
maxillary sinus. No maxillary sinus.
with pus
CT was done.
discharge.

Pain and swelling

Waters view of a lytic Lytic lesion destroying Complete excision through the
in the right
Cakur et al. 23 lesion in the right and displacing the roof Caldwell-Luc approach. No
maxillary region
(2008) [4] years/male maxillary sinus with and lateral wall of the recurrence was reported after six
accompanied by
an impacted tooth. right maxillary sinus. months of follow-up.
pus discharge.

Unilocular
Diffuse painful radiolucency
extra oral swelling extending from the
extending from left root apex of the upper
ala of the nose to right canine to the
Surgical enucleation followed by
Bhagavandas left zygomatic mesial root of the
32 chemical cauterization. Follow up
Rai et al. prominence, intra- upper left first molar. No data available.
years/male for one year and no evidence of
(2010) [2] oral draining sinus There was an inverted
recurrence.
in relation to conical
maxillary left supernumerary tooth

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 second premolar between the left
region. central and lateral
incisors.

Diffuse bilateral Complete destruction of

Surgical decompression after
maxillary swelling the maxillae, palate,
shielding adjacent vital structures
of three years nasal septum, and nasal
with Sofra-Tulle dressing and
involving the right bone by a mass
chemical cautery of the tumour
infraorbital region occupying the whole of
Okoje- bed was achieved with Carnoy’s
38 resulting in the maxillae and
Adesomoju et No data available. solution. The cavities were then
years/male epiphora. Growth maxillary antrum.
al. (2014) [8] packed with argotone-soaked
of both swellings Destruction of the floor of
gauze to allow for proper
resulted in the the orbit, ethmoid,
haemostasis; this was left in
mobility of teeth sphenoid, and frontal
place for three days. The patient
and pus discharge sinuses as well as the
was lost to follow-up.
from 15. roof of the frontal sinus.

Pain and extraoral

swelling on the
Hyperdense area
left middle third
obliterating complete left
region of the face,
Haziness in both the maxillary sinus causing Enucleation followed by
intra-orally distal
Newaskar et 21 right and left maxillary lateral nasal wall at the peripheral ostectomy. No
to the last tooth in
al. (2016) [9] years/male sinus and impacted level of the middle recurrence post two years of
the left upper
teeth 18. concha was destructed follow-up.
back tooth region.
and showed displaced
The swelling was
and impacted tooth 28.
accompanied by
pus discharge.

A well-defined
multilocular
Painless swelling radiolucent lesion with Two well-circumscribed
on the right side of a sclerotic border lesions present in the
Both the lesions were enucleated
the face for the present in the right anterior maxilla and the
with the maxillary sinus, were
Maruthamuthu past six months maxilla extending maxillary sinus region.
45 decompressed and then
et al. (2017) with watering of between right 16 and Deviation of nasal
years/female enucleated. It was a recurrent
[10] the eyes for one 23 mesiodistally and septum towards the left
case but post-operative periodic
month. History of superiorly involving side with obstruction of
follow was done.
surgery at the the right orbit with the right osteomeatal
same site in 2005. discontinuity in the complex.
right infraorbital
margin.

Tooth displaced to the Radiolucent and

Maxillary swelling
orbital floor and multilocular images with Enucleation with curettage, five
Bastos et al. 15 with no
presence of tooth displacement in years follow-up data with no
(2019) [11] years/male associated
radiolucency in right proximity to the floor of recurrence.
symptoms.
maxillae. the right orbit.

Pain, swelling,
and pus discharge Ill-defined, radiolucent
Single large destructive
with respect to the lesion associated with Enucleation along with tooth
Sheethal et al. 15 lesion involving lateral
left 26 and 27 an impacted third number 28 removal. No follow-up
(2019) [12] years/female and posterior wall of the
regions for three molar displaced to the data.
maxillary sinus.
months. Missing left maxillary sinus.
28.

A dense cystic lesion that

No clinical
entirely occupied the
symptoms or any Upper right third
right maxillary sinus Enucleation without the use of
Goto et al. 21 extraoral or intra- molar which was
extending to the right Carnoy’s solution 20 months
(2020) [13] years/male oral swelling displaced into the
pterygoid process of the after the initial surgery.
associated with maxillary sinus.
sphenoid bone near the
the lesion.
skull base.

Right side retro- Cystic mass in right

orbital pain for maxillary sinus and
Walsh et al. 29 four months and An ectopic tooth in the opacification of the Marsupialization post six months

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 (2022) [14] years/female loose upper right right maxillary sinus. maxillary and ethmoidal follow up. No recurrence.
molar. air cells.

TABLE 1: Tabulation of the literature on invasive maxillary sinus odontogenic keratocyst (OKC)
case reports chronologically from 2001 to 2022

The case reports of maxillary sinus OKC compiled in the table including ours were all non-syndromic. OKCs
cover a wide age range, from the first decade of life to as late as the ninth decade [2,3]. The peak occurrence
is seen in the second and third decades, which was similar to the reported case [3,6].

In the current case, the cyst extended into the floor of the orbit, hard plate, and pterygoid plates similar to
the case reported by Goto et al. [13]. The majority of the maxillary sinus OKCs reported were symptomatic
although this case was asymptomatic [2,4,7-9]. As per the literature, 25% to 40% of the cases
have involvement of an unerupted impacted, displaced, or incompletely formed tooth [2,4,7-14]. In the
reported case, there was no association with either any impacted or undeveloped tooth or any discharge
associated with the lesion.

OKCs have a multifactorial theory of origin. Initially, the primordium of the tooth was thought by many
authors to be the origin of these cysts and hence the name primordial cyst but now the dental lamina is
considered to be the most likely origin. The basal cell layer of the oral epithelium is also thought to possibly
play a role in the aetiology of these cysts [3]. The origin of OKC in the maxillary sinus is controversial,
presumably arising from the entrapment of odontogenic epithelium within the sinus because of the close
anatomy. A breach in the Schneiderian membrane due to odontogenic infection or odontogenic pathology of
the maxillary bone can lead to maxillary sinus infection [14].

The theory of intrinsic growth potential of a cyst due to expression of Ki67, proliferating cell nuclear antigen
(PCNA), and p-63 has also been proposed according to which patients who have the predisposition to form
keratocyst will always have a higher risk of developing a cyst as long as a dental lamina or its remnant are
present [15]. As per studies, Ki67 labelling is higher in cases with PTCH1 mutations. The theory specifically
can be applied to syndromic patients (mutation rate > 85%) but sporadic OKC cases cannot be excluded
(mutation rate < 30%). In the case of sporadic cases where there is epithelium separation from the
connective tissue of the cyst, the PTCH1 mutation rate increases to 84% nearly equal to the syndromic
OKC [16].

This theory could also be applied to the current case considering its behaviour and invasive potential but
confirmation required evidence through genomic testing and immunohistochemistry (IHC) expression,
which could not be done due to limited resources. Also, in the reported case, the cyst could have developed
as a result of entrapment and proliferation of odontogenic epithelial cells or extensions of the basal cell
layer of the epithelium of oral mucosa in the sinus. The patient had very poor oral hygiene and severe
periodontitis, which could have additionally attributed to the infection of the sinus.

Several theories have been proposed for explaining the invasive and destructive nature of OKC. Growth in
OKC is linked to unknown growth factors inherent in the epithelium itself or enzymatic activity in the
fibrous wall [15]. Its invasion and infiltration are attributed to the multicentric growth potential that is
cystic growth brought about by the proliferation of local groups of epithelial cells [17]. In the current case,
the cyst was unilocular and there was no multicellularity seen in histopathology yet it was large, expansile,
destroying the sinus floor, and perforating the cortical bone.

Aggressive maxillary sinus OKCs tend to penetrate into the surrounding soft tissues with expansion and
perforation of cortical bone. The expansion has been reported to occur in up to 60% of cases, similar to the
current case reported [3]. Advanced imaging techniques like CT and MRI are more useful as sometimes the
pan tomographs can be misleading in viewing large lesions involving the maxillary sinus and those invading
the skull base or surrounding spaces [13,18]. All case reports of sinus involvement used CT as their chief
source of investigation as it not only demarcates the clear boundaries and extent of the lesion but also helps
to demonstrate other features of OKCs, such as bony changes (expansion in buccolingual/palatal direction
and erosion), internal density, and extension into soft tissue [19]. It also provides a better aid for the
surgeons to prepare their procedures pre-operatively. Even in our case, OPG and the paranasal view did not
give a clear interpretation of the lesion while CT demarcated the lesion boundaries discretely.

There is no universally accepted treatment for OKC. Considering its aggressive nature and history of
recurrence, the primary aim of treatment is to achieve total eradication. The techniques involve
decompression followed by enucleation and peripheral osteotomy, which show less recurrence, as compared
to only enucleation, which has a recurrence rate of 17-56% [13]. The application of Carnoy’s solution has
been very effective in adjunct to peripheral osteotomy and enucleation in extensive lesions in reducing the
recurrence rate [9]. For our case too, freshly prepared Carnoy’s solution was used post-enucleation and

2023 Singh et al. Cureus 15(4): e37357. DOI 10.7759/cureus.37357 10 of 11

 curettage. Even with Carnoy’s solution application, a recurrence rate of 1-8.7% is reported and hence
patient has been kept under periodic follow-up [13].

Conclusions
Maxillary sinus OKCs are less in occurrence and every case has a varied presentation. FNAC is of limited help
and CT imaging should be considered as the baseline radiological investigation to diagnose and plan
treatment. Histopathology, genomic testing, and IHC should be considered as diagnostic criteria as they add
better explanatory data on the aetiology and behaviour patterns of OKCs. Post-operative periodic follow-up
should be mandatory irrespective of the operative procedure followed.

Additional Information
Disclosures
Human subjects: Consent was obtained or waived by all participants in this study. Conflicts of interest: In
compliance with the ICMJE uniform disclosure form, all authors declare the following: Payment/services
info: All authors have declared that no financial support was received from any organization for the
submitted work. Financial relationships: All authors have declared that they have no financial
relationships at present or within the previous three years with any organizations that might have an
interest in the submitted work. Other relationships: All authors have declared that there are no other
relationships or activities that could appear to have influenced the submitted work.

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