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 Table of Contents  
ORIGINAL ARTICLE
Year : 2016  |  Volume : 4  |  Issue : 1  |  Page : 40-44

Oral mucocele: A clinicopathological analysis of 50 cases


1 Department of Oral Pathology, Faculty of Dentistry, SEGi University Kota Damansara, Selangor, Malaysia
2 Department of Oral and Maxillofacial Pathology, Hitkarini Dental College and Hospital, Jabalpur, Madhya Pradesh, India
3 Department of Pediatric Dentistry, Faculty of Dentistry, SEGi University Kota Damansara, Selangor, Malaysia

Date of Web Publication2-Jun-2016

Correspondence Address:
Anand S Tegginamani
Department of Oral Pathology, Faculty of Dentistry, SEGi University Kota Damansara, PJU 5, 47810 Petaling Jaya, Selangor
Malaysia
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Source of Support: None, Conflict of Interest: None


DOI: 10.4103/2321-4848.183357

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  Abstract 

Background: Mucocele belongs to the category of reactive lesions affecting the salivary gland related to obstruction or trauma of the salivary glands. These are pseudocysts formed due to the accumulation of pooled mucus. Objectives: To evaluate the clinicopathologic features including variations of 50 oral mucocele cases Materials and Methods: Archival review for the 50 cases reported as mucoceles was performed, and data for the following parameters were recorded: Age, gender, history of trauma, site and clinical presentation, variation in histology, treatment mode, and recurrence. Results: A total of 50 cases of mucoceles were retrieved for the archives for study. All were extravasation phenomenon (100%), no retention phenomenon was found, seen in the second (36%) and third (46%) decade, with male preponderance (male:female 19:6). Most common locations were lower lip (96%), floor of the mouth (6%), and ventral tongue (4%). Commonly presented as soft, fluctuant, bluish swelling associated with history of trauma. Variations included superficial mucocele (10%), clear cell change (4%) and papillary infoldings (2%) and 6% of recurrence cases. Conclusion: Although it is a benign lesion which is commonly encountered and excised in dental practice, the classic clinical presentation of soft, fluctuant swelling accompanied by history of evolution of the lesion does not pose much difficulty in diagnosis. Special variants of oral mucoceles occur infrequently; it is important to recognize these variants to avoid misdiagnosis, it is always wise to subject it to histopathological confirmation owing to its close clinical resemblance to neoplastic condition.

Keywords: Clear cell variants, papillary projections, rhabdomyosarcoma, superficial mucocele, swellings of lower/upper lip


How to cite this article:
Tegginamani AS, Sonalika WG, Vanishree H S. Oral mucocele: A clinicopathological analysis of 50 cases. Arch Med Health Sci 2016;4:40-4

How to cite this URL:
Tegginamani AS, Sonalika WG, Vanishree H S. Oral mucocele: A clinicopathological analysis of 50 cases. Arch Med Health Sci [serial online] 2016 [cited 2019 Dec 14];4:40-4. Available from: http://www.amhsjournal.org/text.asp?2016/4/1/40/183357


  Introduction Top


Mucocele belongs to the category of reactive lesions affecting the salivary gland related to obstruction or trauma of the salivary glands. These are pseudocysts formed due to the accumulation of pooled mucus. Histologically, they are categorized into two types: Mucus extravasations phenomenon and mucus retention phenomenon. Extravasation phenomenon commonly affects minor salivary glands (mSGs), especially of the lower lip and seen commonly in children and young adults. In contrast to this, mucus retention phenomenon is seen in older age group, and major salivary glands are more frequently involved.[1] The lesion can be located directly under the mucosa (superficial mucocele), in the upper submucosa (classic mucocele), or in the deep corium (deep mucocele).[2]

The classic clinical presentation of soft, fluctuant swelling accompanied by history of evolution of the lesion does not pose much difficulty in diagnosis. Sometimes lesions such as vesiculobullous lesions, hemangioma, and neoplastic diseases such as mucoepidermoid carcinoma closely mimic mucocele and this fact warrants histopathological examination of all the excised mucocele.[1],[3] Variation in mucocele has also been documented [4] which broadens the differential diagnosis. Thus, a retrospective study was conducted to characterize the clinicopathological details of oral mucocele and to as certain types and frequency of variations in its presentation.


  Materials and Methods Top


Departmental archives were retrieved for retrospective analysis of cases reported as mucoceles. Clinical data under the following parameters were recorded: Age, sex, and site, history of trauma, presentation, treatment mode, and recurrences. Histological features were evaluated by two oral pathologists for type: Extravasation/retention phenomenon and variations present.


  Results and Observations Top


A total of 50 cases of mucoceles were retrieved. Males showed a distinct predominance [Figure 1], the male:female ratio being 19:6. Age of the patient ranged from 11 to 53 years with a mean age of 25.24 years. Maximum cases were seen in the second (36%) and third decades (46%) [Figure 2]. Most commonly lower lip was involved (90%) followed by the floor of the mouth (6%) and ventral surface of the tongue (4%) [Figure 3]. Lesion manifested as a bluish translucent swelling which was nontender and fluctuant (60%), nodular swellings with normal mucosal color (36%), and ulceration (4%). History of trauma was present in 76% of the cases, mostly lip biting. Histologically, all belonged to extravasation type characterized by mucous pooling with wall of granulation tissue. Two cases were in organizing stage [Figure 4]. Sixteen percent of mucoceles showed variations such as superficial mucoceles (10%) [Figure 5], extensive clear cell change (4%) [Figure 6], and papillary projections into the lumen covered by granulation tissue (2%) [Figure 7] were noted. Surrounding salivary gland showed features such as sialadenitis, atrophy, and ductal dilation. No retention cyst was present. All cases underwent surgical excision of the lesion along with lobules of adjacent mSGs. Six percent cases showed recurrence.
Figure 1: Graphical representation for gender distribution of mucoceles

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Figure 2: Graphical representation depicting frequency of occurrence of mucoceles in different decades

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Figure 3: Pie chart showing site-wise distribution of mucoceles

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Figure 4: Mucocele in organizing stage with obliteration of cystic space by growth of granulation tissue (H and E, ×4)

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Figure 5: Superficial mucocele with mucin spillage immediately beneath the epithelium which is elevated and thinned (H and E, ×4)

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Figure 6: Extensive clear cell change in mucocele (H and E, ×10)

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Figure 7: Papillary projection covered by granulation tissue wall into the cystic space (H and E, ×10)

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  Discussion Top


Obstructive disorders are a relatively common disorder affecting the major and mSGs. The causative mechanism underneath being reduction or cessation of salivary flow which is attributed to one of the two processes: Destruction of secretory elements of the gland or a mechanical blockage or disruption of the ductal system that leads from the gland to the oral cavity.[5] The latter process being far more common leading to the pathological entity entitled “mucoceles.” It contributes to the largest number of nonneoplastic salivary gland disorders. Based on the pathogenic process, mucoceles are classified into two categories: “extravasation mucocele” being defined as pooling of mucus in a cavity within the connective tissue that is not lined by epithelium, and the ones which result due to obstruction of duct leading to true cysts lined by epithelium are termed as “retention mucoceles.”[1],[5]

Lower lip (90%) was the most frequent location followed by the floor of the mouth (6%) and ventral surface of the tongue (4%). Mucous extravasation phenomenon is commonly noted in the lower lip as a consequence of traumatic injury to the labial mSG due to lip biting.[6],[7] Gender predilection varies from male predominance [7] or female predominance [8] to no gender predilection.[6] Our study showed a definitive male preponderance. Although it can occur in any age group, it is frequently encountered in children and young adults.[5] We noted the maximum distribution in the second (36%) and third decade (46%). In contrast to the young age and predominant involvement of mSGs in extravasation phenomenon, the retention phenomenon affects the major salivary glands and in older age groups.[1] In our series, no retention phenomenon was found.

The clinical presentation depends on the depth of the location of lesion within the soft tissue [5] and also the extensibility of the surrounding connective tissue.[1] Superficial lesions manifest as soft tissue swelling which are vesicular, translucent and bluish in appearance, and fluctuant on palpation. The vesicle may rupture leading to ulceration.[3],[5] Such presentation should be carefully differentiated from vesiculobullous lesions of the oral cavity.[1],[3],[5] The presence of mSG tissue, overlying normal surface epithelium, and positive mucicarmine staining facilitate appropriate diagnosis of mucocele.[9] Deeper lesions present as nodular swelling which is of normal mucosal color.[5]

Histologically, mucous pooling walled by granulation tissue containing numerous mucinophages is characteristic, compressing the surrounding connective tissue. Salivary gland acini or ductal element can be frequently seen adjacent to the lesion.[5],[10] As the lesion ages, the cavity is obliterated with the progressive growth of granulation which is then termed as “organizing mucocele”[5] [Figure 4]. The adjacent salivary gland may also show changes including sialadenitis, ductal dilation, and atrophy of acini.[10] The lesion may stretch the overlying epithelium causing thinning and flattening of rete ridges [Figure 4] and [Figure 5]. Histopathologist has to be meticulous keeping in mind the close resembles of this lesion to low-grade mucoepidermoid carcinoma which is also featured by cystic spaces, mucin pools, and presence of mucin-filled mucocytes. However, the absence of epithelial lining favors mucocele.[5] Extensive clear cell changes in mucoceles can mimic clear cell neoplasm which requires detailed investigations including histochemistry, and immunohistochemistry.[11] Apart from this, variations such as superficial mucoceles, myxoglobulosis, and papillary synovial metaplasia-like changes have been reported;[4] later, two variations are thought to be histogenetically interrelated [12] representing healing response under special circumstances.[13] This variation is seen, especially in lower lip mucocele, as a consequence to repeated traumatic episodes [12] which disrupts the classic process of healing by granulation tissue and brings in this alternate pathway.[14] In our series, we noticed papillary projections in two of our cases, covered by tissue consisting of spindled fibroblasts and epithelioid macrophages, but typical synovial metaplasia-like changes comprising polarized fibriohistocytic cells oriented parallel to the lumen [13] were not evident. These two cases might represent very early stages of these mentioned variations but can be stated only after histological evidence regarding their evolutionary stage which requires a study on the larger number of these cases.

Recently Keshet et al. reported that superficial mucoceles in cancer patient after radiation therapy and proposed theory that radiation therapy along with chemotherapy (Cisplatin) mediated damage to mSGs ducts and aquaporins as a reason for superficial oral mucocele formation in cancer patient after radiation therapy. Contrary to that, being close to the oral mucosa, mSGs, especially their excretory ducts, are also exposed to the carcinogenic attack of detrimental oral habits. Unlike oral epithelium, such carcinogenic attack can mediate disruption of basement membrane and loss of cohesion in the ductal components of mSGs. This could lead to leakage in the ductal system causing spillage of mucous secretions in the adjacent connective tissue and subsequent superficial mucocele formation and showed evidences of such lesions at surgical margins before chemo-radio therapy, the possibility of carcinogen-mediated damage to the ducts of mSGs, as causative factor for “postchemoradiotherapy” related superficial mucocele cannot be ruled out. Oral squamous cell carcinoma is preceded by myriad of potentially malignant disorders, which are associated with various kinds of carcinogenic insults to the oral mucosa. Each disorder has potential to affect the mSGs present underneath the mucosa.[14],[15]

Surgical removal of the lesion along with adjacent lobules of mSG and duct is most widely practiced. Superficial extravasation mucocele resolves spontaneously and requires no treatment. Recurrences, although low, are result of residual salivary gland requiring removal of the lesion down up to the muscle layer. Placement of suture after excision should also be very carefully executed to avoid injury to the adjacent glands and ducts which can cause recurrence.[14],[16],[17]


  Conclusion Top


Mucoceles are mostly benign and self-limiting nature, primarily diagnosed based on clinical findings followed by definitive diagnosis based on the histopathological investigation. Most of the reported literature showed lesion arose followed by trauma and habitual lip biting. Hence, a school-based educational awareness program for both children and parent at a 6 months interval and interception of the oral habit among children is the key factor. Patient undergoing orthodontic therapy should be monitored periodically for areas of irritation in the oral mucosa. Findings of this study are in concordance with earlier studies that extravasation phenomenon is far more common than retention and extravasation mucocele showed a definite male predominance which most frequently noticed in the second and third decade of life. Although it is a benign lesion which is commonly encountered and excised in dental practice, the classic clinical presentation of soft, fluctuant swelling accompanied by the history of evolution of the lesion does not pose much difficulty in diagnosis. Special variants of oral mucoceles occur infrequently, it is important to recognize these variants to avoid misdiagnosis, it is always wise to subject it to histopathological confirmation owing to its close resemblance to neoplastic, lesions such as vesiculobullous lesions, hemangioma, and neoplastic diseases such as mucoepidermoid carcinoma, embryonal rhabdomyosarcoma of lower lip closely mimic mucocele, and this fact warrants histopathological examination of all the excised mucoceles.[1],[3],[18],[19],[20],[21]

Financial support and sponsorship

Nil.

Conflicts of interest

There are no conflicts of interest.

 
  References Top

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Conceição JG, Gurgel CA, Ramos EA, De Aquino Xavier FC, Schlaepfer-Sales CB, Cangussu MC, et al. Oral mucoceles: A clinical, histopathological and immunohistochemical study. Acta Histochem 2014;116:40-7.  Back to cited text no. 8
    
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Hayashida AM, Zerbinatti DC, Balducci I, Cabral LA, Almeida JD. Mucus extravasation and retention phenomena: A 24-year study. BMC Oral Health 2010;10:15.  Back to cited text no. 9
    
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Piña AR, Almeida LY, Andrade BA, León JE. Clear cell change in a lower lip mucocele. J Oral Maxillofac Pathol 2013;17:318.  Back to cited text no. 11
    
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Ide F, Kikuchi K, Kusama K. Is papillary synovial metaplasia-like change a hitherto undescribed histologic pattern of lip mucoceles? Oral Surg Oral Med Oral Pathol Oral Radiol Endod 2010;109:800-1.  Back to cited text no. 12
    
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Krishnanandan S, Abbassian A, Sharma AK, Cunnick G. Capsular synovial metaplasia mimicking silicone leak of a breast prosthesis: A case report. J Med Case Rep 2008;2:277.  Back to cited text no. 13
    
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Keshet N, Abu-Tair J, Zaharia B, Abdalla-Aslan R, Aframian DJ, Zadik Y. Superficial oral mucoceles in cancer patient after radiation therapy: An overlooked yet imperative phenomenon. Oral Oncol 2016;52:e1-2.  Back to cited text no. 14
    
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Sarode SG, Sarode CS. Superficial oral mucoceles in cancer patient after radiation therapy: An overlooked yet imperative phenomenon. Letter to editor. Oral Oncol 2016;56:1-2. [Epub ahead of print].  Back to cited text no. 15
    
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Huang IY, Chen CM, Kao YH, Worthington P. Treatment of mucocele of the lower lip with carbon dioxide laser. J Oral Maxillofac Surg 2007;65:855-8.  Back to cited text no. 16
    
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Ata-Ali J, Carrillo C, Bonet C, Balaguer J, Penarrocha M, Penarrocha M. Oral mucocele: Review of the literature. J Clin Exp Dent 2010;2:e10-3.  Back to cited text no. 17
    
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Seth T, Kempert P. Embryonal rhabdomyosarcoma of lower lip. Indian Pediatr 2004;41:858-9.  Back to cited text no. 18
    
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D'Amico AV, Goldwein J, Womer R. Alveolar rhabdomyosarcoma of the lip in an infant. Med Pediatr Oncol 1996;26:409-13.  Back to cited text no. 19
    
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Nallasivam KU, Sudha BR. Oral mucocele: Review of literature and a case report. J Pharm Bioallied Sci 2015;7 Suppl 2:S731-3.  Back to cited text no. 20
    
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Chi AC, Lambert PR 3rd, Richardson MS, Neville BW. Oral mucoceles: A clinicopathologic review of 1,824 cases, including unusual variants. J Oral Maxillofac Surg 2011;69:1086-93.  Back to cited text no. 21
    


    Figures

  [Figure 1], [Figure 2], [Figure 3], [Figure 4], [Figure 5], [Figure 6], [Figure 7]


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