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CASE REPORT
Year : 2018  |  Volume : 36  |  Issue : 3  |  Page : 315-318
 

Blandin and Nuhn mucocele in a pediatric patient


Department of Pedodontics and Preventive Dentistry, Mar Baselios Dental College, Kothamangalam, Kerala, India

Date of Web Publication24-Sep-2018

Correspondence Address:
Dr. Sherin C Jose
Department of Pedodontics and Preventive Dentistry, Mar Baselios Dental College, Kothamangalam, Kerala
India
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Source of Support: None, Conflict of Interest: None


DOI: 10.4103/JISPPD.JISPPD_10_18

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   Abstract 


Oral mucoceles are benign lesions that may appear in any location on the mucosal surfaces of the oral cavity where underlying minor salivary glands are found. The lower lip is the most frequently affected and the most widely reported location. Mucoceles involving the glands of Blandin and Nuhn are infrequent and constitute only about 1.9%–10.3% of the reported cases. Superficial mucoceles are a rare subtype of the Blandin and Nuhn mucoceles and are found only in 4.3%–10% of the mucocele excisions. The purpose of this report is to present a case of a superficial mucocele in the ventral surface of the tongue in a 7-year-old child. The lesion was treated with excisional biopsy.


Keywords: Mucocele, salivary gland, tongue


How to cite this article:
Jose SC, Abraham K K, Khosla E. Blandin and Nuhn mucocele in a pediatric patient. J Indian Soc Pedod Prev Dent 2018;36:315-8

How to cite this URL:
Jose SC, Abraham K K, Khosla E. Blandin and Nuhn mucocele in a pediatric patient. J Indian Soc Pedod Prev Dent [serial online] 2018 [cited 2019 Nov 14];36:315-8. Available from: http://www.jisppd.com/text.asp?2018/36/3/315/241957





   Introduction Top


Incidences of pathological conditions of the mouth and perioral structures are common in the oral cavity of children.[1] Among the soft-tissue pathology, salivary gland pathology was the most common diagnostic category and 87.5% of diagnoses in this category were mucous extravasation cysts, which was the most commonly diagnosed oral lesion in children.[2]

The mucocele of the oral mucosa usually results from rupture of a salivary gland duct and results in mucin spillage into the surrounding soft tissues. The mucin spillage is often associated with local trauma, even though there is no known history of trauma in some cases.[3] In a series of cases reported by Standish and Shafer, nearly 45% of the mucoceles occurred on the lower lip.[4] This article presents a case report of a 7-year-old child with an unusual representation of Blandin and Nuhn mucocele on the ventral surface of the tongue.


   Case Report Top


A female patient aged 7 years was referred to the department with a chief complaint of a swelling in relation to the ventral surface of the tongue for 3 months. The patient gave a history of the previous visit to a pediatrician for the same, where they have attempted to compress the swelling and noticed a minor reduction in the size. Later, the swelling regained its original size and was hence referred to the dentist.

On examination, a pedunculated growth of size 10 mm × 5 mm was seen on the ventral surface of the tongue. The superficial mucosa was erythematous with irregular surface and was lobulated. It was mildly tender on palpation, and it did not bleed on palpation [Figure 1].
Figure 1: Swelling in the ventral surface of the tongue

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Intraorally, the patient had retained lateral incisors and lingually erupting permanent successors. The growth in the tongue occupied the space between the two erupting permanent lateral incisors. This led to the provisional diagnosis of irritational fibroma and the two mandibular deciduous lateral incisors were extracted and the patient was called for excisional biopsy of the growth.

On the second visit after 3 days, the patient showed a surprising reduction in size of the growth to about 5 mm × 5 mm after the traumatic etiologic factor was removed [Figure 2]. There was also a reduction in the redness of the growth and it appeared more similar to normal mucosa. Conservative approach to the growth was planned, and the patient was followed up for 2 weeks. After 2 weeks, no considerable change in the size of the growth was noticed, and hence, a circumferential excisional biopsy was performed under local anesthesia and the specimen was subjected to microscopic analysis.
Figure 2: Swelling reduced in size after the retained lateral incisors were removed

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Histopathological examination showed keratinized stratified squamous epithelium in association with fibrovascular connective tissue. The subepithelial connective tissue exhibits areas of mucin spillage and moderately dense chronic inflammatory cells. The section also showed mucous minor salivary gland acini and dilated ducts [Figure 3] and [Figure 4]. Deeper areas also showed muscle fibers, vascular spaces, and extravasated RBC's. The histopathological features were suggestive of superficial mucocele.
Figure 3: (1) Minor salivary gland acini. (2) Ducts of minor salivary gland

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Figure 4: (3) Stratified squamous surface epithelium. (4) Subepithelial split. (5) Mucin spillage

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Follow-up was done at 3 months interval for 1 year and 6 months interval for the next year and showed no signs of recurrence.


   Discussion Top


The mucocele is a salivary gland lesion that originates when the main duct of the minor salivary gland is torn due to trauma and results in mucus extravasation into the connective tissue.[5] Unlike the salivary duct cyst, the mucocele is not a true cyst as it lacks an epithelial lining.[3]

They are common in children and young adults because younger people are more likely to experience trauma that induces mucin spillage. However, mucoceles have been reported in patients of all ages, including newborn infants and older people. The lower lip is the most common site for the mucocele, accounting for over 60% of all cases. Less common sites include the buccal mucosa, anterior ventral tongue, and floor of mouth (ranula).[3],[4]

Mucoceles that arise on the ventral surface of the tongue, situated on both sides of the midline, are known as mucocele of the glands of Blandin and Nuhn. They are embedded in the muscles of the ventral aspect and re-covered by a thin layer of mucosa.[6],[7],[8]

The superficial mucocele is a variant of the extravasation type mucocele and is located under the mucous membrane whereas classical mucoceles are seen in the upper submucosa.[9] Its location as suggested by Jensen was attributed to the fact that increased pressure caused by mucous plugs in the intraepithelial squamous cell lined portion of the duct might cause the duct to rupture, creating superficial mucoceles. This also explains the close proximity of subepithelial blisters to the salivary gland and ducts that is characteristic of these mucoceles.[10]

Ordinary mucoceles are more common in patients <30 years of age, in both men and women, whereas superficial mucoceles are found more frequently in patients over the age of 30 and are found commonly in women.[11] These lesions are asymptomatic and numerous, occurring most often in the retromolar area, soft palate, and posterior buccal mucosa.[12] Their etiology may be closely related to the mechanism and tartar control toothpaste may be a contributory factor in the onset.[13]

Superficial mucoceles are a rare subtype and are found in only 4.3%–16% of mucocele excisions.[14],[15] The true incidence of superficial mucoceles is difficult to assess because some lesions spontaneously resolve, or are short-lived, or are asymptomatic and thus affected individuals might not seek medical attention.

Histopathologically, superficial mucoceles featured a dome-shaped, thin epithelial roof with extravasated mucin directly beneath and admixed with granulation tissue and composed of a lamina propria with a mild inflammatory cell infiltrate.[16]

If the clinician does not directly observe the eruptions or is unaware of their existence, this disease can be easily misdiagnosed. The most common misdiagnoses are bullous lichen planus and mucous membrane pemphigoid. In bullous lichen planus, the bullae are usually large, flaccid, and opaque, when compared to the translucent and tense vesicle of mucocele. The blister of a mucous membrane pemphigoid is easily ruptured and tends to extend at the periphery.[17]

Several modalities of treatment have been instituted for superficial mucoceles, the most likely being surgical excision. For the management of moderate-to-large Blandin–Nuhn mucoceles, Baurmash suggested to completely unroof the lesion along its entire periphery to visualize and remove all of the glands present and complete excision for smaller ones.[18]

Other treatment options include cryosurgery,[19] diode laser,[20] CO2 laser ablation,[21] intralesional corticosteroid injections,[22],[23] micromarsupialization, marsupialization, and electrocautery. The use of alginate impression material to define the outline of the lesion in treating an anterior lingual salivary gland mucocele has also been reported.[24]


   Conclusion Top


Mucoceles of the glands of Blandin and Nuhn are considered to be rare. According to Jinbu et al., Blandin–Nuhn mucoceles comprise 9.9% of all oral mucoceles studied by them.[6] Mucoceles of the glands of Blandin and Nuhn are self-limiting, and with excision including the margins of the swelling and by avoiding future trauma to the site, the chances of recurrence of the swelling are minimized.

Declaration of patient consent

The authors certify that they have obtained all appropriate patient consent forms. In the form the patient(s) has/have given his/her/their consent for his/her/their images and other clinical information to be reported in the journal. The patients understand that their names and initials will not be published and due efforts will be made to conceal their identity, but anonymity cannot be guaranteed.

Financial support and sponsorship

Nil.

Conflicts of interest

There are no conflicts of interest.



 
   References Top

1.
Welbury R, Duggal MS, Hosey MT, editors. Paediatric Dentistry. United Kingdom: Oxford University Press; 2012. p. 313.  Back to cited text no. 1
    
2.
Brierley DJ, Chee CK, Speight PM. A review of paediatric oral and maxillofacial pathology. Int J Paediatr Dent 2013;23:319-29.  Back to cited text no. 2
    
3.
Neville BW, Damm DD, Chi AC, Allen CM. Oral and Maxillofacial Pathology. Philadelphia: Elsevier Health Sciences; 2015. p. 389-91.  Back to cited text no. 3
    
4.
Rajendran R. Shafer's Textbook of Oral Pathology. India: Elsevier; 2012. p. 2163-8.  Back to cited text no. 4
    
5.
Dean JA, Avery DR, McDonald RE. McDonald and Avery Dentistry for the Child and Adolescent-E-Book. Missouri: Elsevier Health Sciences; 2010. p.681  Back to cited text no. 5
    
6.
Jinbu Y, Kusama M, Itoh H, Matsumoto K, Wang J, Noguchi T, et al. Mucocele of the glands of Blandin-Nuhn: Clinical and histopathologic analysis of 26 cases. Oral Surg Oral Med Oral Pathol Oral Radiol Endod 2003;95:467-70.  Back to cited text no. 6
    
7.
Sugerman PB, Savage NW, Young WG. Mucocele of the anterior lingual salivary glands (glands of Blandin and Nuhn): Report of 5 cases. Oral Surg Oral Med Oral Pathol Oral Radiol Endod 2000;90:478-82.  Back to cited text no. 7
    
8.
Ellis E 3rd, Scott R, Upton LG. An unusual complication after excision of a recurrent mucocele of the anterior lingual gland. Oral Surg Oral Med Oral Pathol 1983;56:467-71.  Back to cited text no. 8
    
9.
Rao PK, Ram SS, Laxmikanth C, Prashanth S. Oral mucocele – A mini review. Dentistry 2013;3:15.  Back to cited text no. 9
    
10.
Jensen JL. Superficial mucoceles of the oral mucosa. Am J Dermatopathol 1990;12:88-92.  Back to cited text no. 10
    
11.
Bermejo A, Aguirre JM, López P, Saez MR. Superficial mucocele: Report of 4 cases. Oral Surg Oral Med Oral Pathol Oral Radiol Endod 1999;88:469-72.  Back to cited text no. 11
    
12.
Regezi JA, Sciubba JJ, Jordan RC. Oral Pathology: Clinical Pathologic Correlations. Missouri: Elsevier Health Sciences; 2003. p. 183-5.  Back to cited text no. 12
    
13.
Navazesh M. Tartar-control toothpaste as a possible contributory factor in the onset of superficial mucocele: A case report. Spec Care Dentist 1995;15:74-8.  Back to cited text no. 13
    
14.
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. 14
    
15.
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. 15
    
16.
Brooks JK, Schwartz KG, Basile JR. Superficial mucocele of the ventral tongue: Presentation of a rare case and literature review. J Oral Maxillofac Surg 2016;74:1175-9.  Back to cited text no. 16
    
17.
Inoue A, Ikeda S, Mizuno Y, Ogawa H. Superficial mucoceles of the soft palate. Dermatology 2005;210:360-2.  Back to cited text no. 17
    
18.
Baurmash HD. Mucoceles and ranulas. J Oral Maxillofac Surg 2003;61:369-78.  Back to cited text no. 18
    
19.
Twetman S, Isaksson S. Cryosurgical treatment of mucocele in children. Am J Dent 1990;3:175-6.  Back to cited text no. 19
    
20.
Paglia M, Crippa R, Ferrante F, Angiero F. Mucocele of the minor salivary glands in an infant: Treatment with diode laser. Eur J Paediatr Dent 2015;16:139-42.  Back to cited text no. 20
    
21.
Kopp WK, St-Hilaire H. Mucosal preservation in the treatment of mucocele with CO2 laser. J Oral Maxillofac Surg 2004;62:1559-61.  Back to cited text no. 21
    
22.
Wilcox JW, History JE. Nonsurgical resolution of mucoceles. J Oral Surg 1978;36:478.  Back to cited text no. 22
    
23.
Luiz AC, Hiraki KR, Lemos CA Jr., Hirota SK, Migliari DA. Treatment of painful and recurrent oral mucoceles with a high-potency topical corticosteroid: A case report. J Oral Maxillofac Surg 2008;66:1737-9.  Back to cited text no. 23
    
24.
Kumaresan R, Karthikeyan P, Mohammed F, Thapasum Fairozekhan A. A novel technique for the management of Blandin-Nuhn mucocele: A case report. Int J Clin Pediatr Dent 2013;6:201-4.  Back to cited text no. 24
    


    Figures

  [Figure 1], [Figure 2], [Figure 3], [Figure 4]



 

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