[Downloaded free from http://www.joomr.org on Tuesday, March 14, 2017, IP: 190.92.95.

83]

CaseReport

Idiopathic mucocele of maxillary sinus: A rare

and frequently misdiagnosed entity
Abhilasha Sadhoo, Isha Preet Tuli1, Nishi Sharma1
Department of Head and Neck Oncology, Kidwai Memorial Institute of Oncology, Bengaluru, Karnataka, 1Department of Otorhinolaryngology,
PGIMER and DrRML Hospital, NewDelhi, India

A B S T R A C T

Maxillary sinus mucocele is an unusual uncommon benign lesion which develops due to retained secretions and presents as an
expansile cystic lesion. Presenting symptoms are nonspecific and mostly due to pressure effects on the orbit or facial deformity.
Etiology of such mucoceles is not well understood, and it is postulated that they ensue due to obstruction of the ostium by inflammation
or previous procedures such as CaldwellLuc surgery. De novo origins are rare causing unnecessary investigations and delayed
diagnosis and treatment. Endoscopic marsupialization of the mucocele is the surgery of choice, even though complicated cases maybe
approached by an external route. We present a case of right maxillary mucocele without an identifiable cause, which is reported due
to its rarity and delayed management.

Key words: Endoscopic surgery, maxillary sinus, mucocele

Introduction of a slow growing, rightsided facial swelling and dull

Mucocoele of the paranasal sinus is an epithelial lined,
mucus containing sac that can fill the sinus completely and is
capable of expansion. The frontoethmoid sinuses(89%) are
the most commonly affected and the maxillary sinus(1%)
the least.[1] They arise consequent to obstruction of the
ostium and inflammation due to previous surgery or trauma
of the paranasal sinuses.[1,2] In onethird of the cases,
mucoceles are idiopathic in origin.[2] Here, we report a case
of right maxillary mucocele with no apparent cause, leading
to its initial misdiagnosis, and thus, delayed management.
Case Report
A 44yearold diabetic male presented to our Ear, Nose,
Throat outpatient department with a 2month history
Access this article online
Quick Response Code:
Website:
www.joomr.org
pain. The swelling had appeared insidiously, was intially
small that gradually grew to its present size, and was
accompanied with a dull localized pain that was not relieved
by medication. There was no history of nasal obstruction
or discharge, loosening of teeth, epiphora, hyposmia or
anosmia, trismus, or fever.
The patient was conservatively treated, initially by a dentist
with oral antibiotics and antiinflammatory drugs. He
was then referred to an otolarynologist who performed
a sublabial biopsy when the symptoms did not abate.
Histopathology report suggested a nonmalignant lesion,
for which he was referred to a higher centre for further
evaluation. The patient was subjected to another transnasal
endoscopic biopsy(details of which are not available), and
This is an open access article distributed under the terms of the
Creative Commons AttributionNonCommercialShareAlike 3.0
License, which allows others to remix, tweak, and build upon the
work noncommercially, as long as the author is credited and the
new creations are licensed under the identical terms.
For reprints contact: reprints@medknow.com

DOI: Cite this article as: Sadhoo A, Tuli IP, Sharma N. Idiopathic mucocele of
10.4103/2321-3841.196360 maxillary sinus: A rare and frequently misdiagnosed entity. J Oral Maxillofac
Radiol 2016;4:87-9.

Address for correspondence: Dr.Isha P. Tuli, Department of Otorhinolaryngology, PGIMER and DrRML Hospital, NewDelhi, India.
Email:ishatuli@yahoo.co.in

2016 Journal of Oral and Maxillofacial Radiology | Published by Wolters Kluwer - Medknow 87
[Downloaded free from http://www.joomr.org on Tuesday, March 14, 2017, IP: 190.92.95.83]
Sadhoo, etal.: Mucocele of maxillary sinus
the histopathology was inconclusive. Thereafter, the patient
reported to us.
Local examination revealed a rightsided, firm, nontender
diffuse swelling 3 3 cm in size, which extended from
the infraorbital region to the angle of mouth and lateral
border of the nose to the zygomatic arch. The surface
temperature was not raised. Arightsided palatal buldge
was present from the right upper lateral incisor anteriorly
to the second premolar posteriorly[Figure1].
Diagnostic nasal endoscopy of the right nasal cavity showed
a blocked osteomeatal complex. The lateral wall of nose
was buldging towards the septum, completely occluding
the right nasal passage. Rest of the otolaryngological,
ophthalmological, dental examinations, and general physical
examination were unremarkable.
A fineneedle aspiration(FNA) from the sublabial route
showed low cellularity with focal collections of acute and
chronic inflammatory cells, cystic macrophages, but no
evidence of neoplastic pathology.
Contrastenhanced computed tomography(CECT) of
the nose and paranasal sinus demonstrated large expansile
homogenous mass with no peripheral enhancement,
involving the right maxillary sinus pushing its medial wall
medially and occluding the right nasal cavity. There was
mucosal thickening with compression of nasal septum and
inferior and middle turbinate[Figure2].
Based on clinical, endoscopic, and radiological features a
provisional diagnosis of a cystic lesion of right maxillay sinus
was made and planned for endoscopic marsupialization and
biopsy under general anesthesia. Rightsided uncinectomy
Figure1: This photograph of the patient shows the rightsided maxillary
swelling (red arrow) and rightsided intraoral bulge(black arrow) taken at
the time of presentation to us
88 Journal of Oral and Maxillofacial Radiology / SeptemberDecember 2016 / Vol 4|Issue 3
was done. The bony buldge of the middle meatal region
was removed and a wide middle meatal antrostomy was
done. Mucoid fluid within the sinus was suctioned, and the
thinwalled cyst was marsupialized. Patient had complete
regression of the cheek swelling within 7days and the palatal
buldge in 10days. Histopathology of the cyst wall confirmed
our diagnosis of mucocele demonstrating an exudate of
neutrophils and macrophages, enmeshed in fibrin, with foci
of hemorrhage and lined with pseudostratified columnar
epithelium. The patient was followed over a period of
6months and is presently asymptomatic.
Discussion
Mucoceles result from an obstruction of the sinus ostia
and drainage pattern, with accumulation of mucus within
the sinus cavity. Continual accumulation causes it to
expand from the pressure. Maxillary sinus mucoceles
are exceptional, with an incidence of 310% worldwide.
They are usually sterile and painless with pain indicating
infection.[1,3] Our patient had sought an early medical
opinion because of pain.
Obstruction of the sinus ostium has been suggested as the
primary etiologic factor. This may be due to a mass lesion,
inflammation and fibrosis, osteoma, fibrous dysplasia,
Pagets disease, malignancy, trauma, or previous surgery.[1,3]
In this case, the patient had no precipitating factor for
the development of mucoceles, which led to unnecessary
biopsies and delayed management.
Expansion occurs through the direct effect of positive
pressure within the mucocele. Local production of bone
resorption factors such as prostaglandins, interleukin1,
Figure2: This CECT of the nose and paranasal sinuses, axial cut demonstrates
the presence of the large, expansile nonenhancing mass involving the right
maxillary sinus and nasal cavity pushing the septum to the left side
[Downloaded free from http://www.joomr.org on Tuesday, March 14, 2017, IP: 190.92.95.83]
Sadhoo, etal.: Mucocele of maxillary sinus
and tumor necrosis factor have also been identified at the
interface between the mucocele and bone. These may cause
intraorbital or intracranial extension.[46]
CT is the preferred imaging modality where mucocele
appears as an expanded, airless sinus filled with
homogeneous material. The walls of the sinus may be
either normal or remodelled, with thickening, thinning
and erosion to various degrees often within the same
sinus. The distinction between a mucocele and a mucous
retention cyst can be made by the presence of air outlining
the upper surface of the retention cyst.[3,7] Hence, a good
radiological examination with a high degree of suspicion
is an invaluable tool for early diagnosis of this condition.
Endonasal endoscopic approach is the preferred treatment
in paranasal sinus mucoceles with the advantages of a
minimally invasive surgery.[2,8] Martel et al. analyzed 58
patients of paranasal sinus mucoceles and found that
recurrence rate was lesser in patients treated endoscopically
(4.8%) than those treated by an external approach
(28.5%).[8] We performed an endoscopic marsupialization
to promote the normal function of secretion and drainage,
prevent damage to normal mucocillary clearance, and avoid
external scar, disturbance of trochlea, and supraorbital
paresthesia.
Thus, we believe that, to evaluate any sinonasal mass, a
thorough clinical, radiological, and endoscopic correlation
is mandatory to reach to an early diagnosis and not suspect
malignancy in the first look. This shall obviate the need for
Journal of Oral and Maxillofacial Radiology / SeptemberDecember 2016 / Vol 4|Issue 3
superfluous invasive procedures and protracted treatment
time, as happened in the reported case.
Financial support and sponsorship
Nil.
Conflicts of interest
There are no conflicts of interest.
References
1. LundVJ. Mucoceles. In: GleesonM, editor. ScottBrowns
Otorhinolaryngology, Head and Neck Surgery. 7thed. London: Hodder
Arnold; 2008. p.15318.
2. CaylakliF, YavuzH, CagiciAC, OzluogluLN. Endoscopic sinus surgery
for maxillary sinus mucoceles. Head Face Med 2006;2:29.
3. TuliIP, PalI, ChakrabortyS, SenguptaS. Persistent deciduous molar as
an etiology for a maxillary sinus mucocele. Indian J Otolaryngol Head
Neck Surg 2011;63(Suppl1):68.
4. JonesNS. Management of the frontal sinus. In: LundVJ, editor.
Cummings OtolaryngologyHead and Neck Surgery. 5thed. Philadelphia:
Mosby Elsevier; 2010. p.783.
5. DiazF, LatchowR, Duvall AJ 3rd, QuickCA, EricksonDL. Mucoceles
with intracranial and extracranial extensions. Report of two cases.
JNeurosurg 1978;48:2848.
6. ZainineR, LoukilI, DhaouadiA, EnnailiM, MediouniA, ChahedH, etal.l.
Ophthalmic complications of nasosinus mucoceles. JFr Ophtalmol
2014;37:938.
7. Eggesb HB, RingertzS, HaanaesOC, Dlvik S, ErichsenA, StirisM,
etal.l. CT and MR imaging of the paranasal sinuses in cystic fibrosis.
Correlation with microbiological and histopathological results. Acta
Radiol 1999;40:15462.
8. MartelMartn M, Gras CabrerizoJR, BotheGonzlez C,
MontserratGiliJR, De JuanDelagoM, MassegurSolenchH. Clinical
analysis and surgical results of 58 paranasal sinus mucoceles. Acta
Otorrinolaringol Esp 2015;66:927.
89