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CASE REPORT
Year : 2016  |  Volume : 4  |  Issue : 3  |  Page : 87-89

Idiopathic mucocele of maxillary sinus: A rare and frequently misdiagnosed entity


1 Department of Head and Neck Oncology, Kidwai Memorial Institute of Oncology, Bengaluru, Karnataka, India
2 Department of Otorhinolaryngology, PGIMER and Dr RML Hospital, New Delhi, India

Date of Web Publication21-Dec-2016

Correspondence Address:
Isha Preet Tuli
Department of Otorhinolaryngology, PGIMER and Dr RML Hospital, New Delhi
India
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Source of Support: None, Conflict of Interest: None


DOI: 10.4103/2321-3841.196360

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  Abstract 

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 Caldwell-Luc 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.

Keywords: Endoscopic surgery, maxillary sinus, mucocele


How to cite this article:
Sadhoo A, Tuli IP, Sharma N. Idiopathic mucocele of maxillary sinus: A rare and frequently misdiagnosed entity. J Oral Maxillofac Radiol 2016;4:87-9

How to cite this URL:
Sadhoo A, Tuli IP, Sharma N. Idiopathic mucocele of maxillary sinus: A rare and frequently misdiagnosed entity. J Oral Maxillofac Radiol [serial online] 2016 [cited 2019 Mar 19];4:87-9. Available from: http://www.joomr.org/text.asp?2016/4/3/87/196360


  Introduction Top


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 one-third 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 Top


A 44-year-old diabetic male presented to our Ear, Nose, Throat outpatient department with a 2-month history of a slow growing, right-sided facial swelling and dull 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 anti-inflammatory 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 the histopathology was inconclusive. Thereafter, the patient reported to us.

Local examination revealed a right-sided, 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. A right-sided palatal buldge was present from the right upper lateral incisor anteriorly to the second premolar posteriorly [Figure 1].
Figure 1: This photograph of the patient shows the right‑sided maxillary swelling (red arrow) and right‑sided intraoral bulge (black arrow) taken at the time of presentation to us

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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 fine-needle 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.

Contrast-enhanced 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 [Figure 2].
Figure 2: This CECT of the nose and paranasal sinuses, axial cut demonstratesthe presence of the large, expansile nonenhancing mass involving the right maxillary sinus and nasal cavity pushing the septum to the left side

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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. Right-sided uncinectomy 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 thin-walled cyst was marsupialized. Patient had complete regression of the cheek swelling within 7 days and the palatal buldge in 10 days. 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 6 months and is presently asymptomatic.


  Discussion Top


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 3-10% 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, Paget's 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, interleukin-1, and tumor necrosis factor have also been identified at the interface between the mucocele and bone. These may cause intraorbital or intracranial extension. [4],[5],[6]

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 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 Top

1.
Lund VJ. Mucoceles. In: Gleeson M, editor. Scott-Brown′s Otorhinolaryngology, Head and Neck Surgery. 7 th ed. London: Hodder Arnold; 2008. p. 1531-8.  Back to cited text no. 1
    
2.
Caylakli F, Yavuz H, Cagici AC, Ozluoglu LN. Endoscopic sinus surgery for maxillary sinus mucoceles. Head Face Med 2006;2:29.  Back to cited text no. 2
    
3.
Tuli IP, Pal I, Chakraborty S, Sengupta S. Persistent deciduous molar as an etiology for a maxillary sinus mucocele. Indian J Otolaryngol Head Neck Surg 2011;63(Suppl 1):6-8.  Back to cited text no. 3
    
4.
Jones NS. Management of the frontal sinus. In: Lund VJ, editor. Cummings Otolaryngology- Head and Neck Surgery. 5 th ed. Philadelphia: Mosby Elsevier; 2010. p. 783.  Back to cited text no. 4
    
5.
Diaz F, Latchow R, Duvall AJ 3 rd , Quick CA, Erickson DL. Mucoceles with intracranial and extracranial extensions. Report of two cases. J Neurosurg 1978;48:284-8.  Back to cited text no. 5
    
6.
Zainine R, Loukil I, Dhaouadi A, Ennaili M, Mediouni A, Chahed H, et al.l. Ophthalmic complications of nasosinus mucoceles. J Fr Ophtalmol 2014;37:93-8.  Back to cited text no. 6
    
7.
Eggesbø HB, Ringertz S, Haanaes OC, Dølvik S, Erichsen A, Stiris M, et al.l. CT and MR imaging of the paranasal sinuses in cystic fibrosis. Correlation with microbiological and histopathological results. Acta Radiol 1999;40:154-62.  Back to cited text no. 7
    
8.
Martel-Martín M, Gras-Cabrerizo JR, Bothe-González C, Montserrat-Gili JR, De Juan-Delago M, Massegur-Solench H. Clinical analysis and surgical results of 58 paranasal sinus mucoceles. Acta Otorrinolaringol Esp 2015;66:92-7.  Back to cited text no. 8
    


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