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CASE REPORT
Year : 2018  |  Volume : 36  |  Issue : 1  |  Page : 93-96
 

Bilateral congenital mucous extravasation phenomenon: A rare case with literature review


1 Department of Dentistry, BPS Government Medical College for Women, Khanpur Kalan, Sonepat, Haryana, India
2 Department of Pathology, BPS Government Medical College for Women, Khanpur Kalan, Sonepat, Haryana, India

Date of Web Publication28-Mar-2018

Correspondence Address:
Dr. Hitesh Chander Mittal
Department of Dentistry, BPS Government Medical College for Women, Khanpur Kalan, Sonepat, Haryana
India
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Source of Support: None, Conflict of Interest: None


DOI: 10.4103/JISPPD.JISPPD_291_16

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   Abstract 

The purpose of this paper is to discuss a rare case of bilateral congenital mucous extravasation phenomenon on buccal mucosa near commissure of mouth. The lesions were noted at birth, subsequently enlarged to interfere with feeding. The lesion frequently ruptured, regressed, and again enlarged to interfere with normal function. The lesions were surgically removed under general anesthesia at 11 months of age and the diagnosis was confirmed by histopathological examination. Postoperative follow-up after 14 months showed no recurrence.


Keywords: Congenital lesion, infant, mucous extravasation phenomenon, oral mucocele


How to cite this article:
Mittal HC, Yadav S, Malik S, Breta M. Bilateral congenital mucous extravasation phenomenon: A rare case with literature review. J Indian Soc Pedod Prev Dent 2018;36:93-6

How to cite this URL:
Mittal HC, Yadav S, Malik S, Breta M. Bilateral congenital mucous extravasation phenomenon: A rare case with literature review. J Indian Soc Pedod Prev Dent [serial online] 2018 [cited 2019 Dec 12];36:93-6. Available from: http://www.jisppd.com/text.asp?2018/36/1/93/228745





   Introduction Top


Mucous extravasation cyst (MEC) or oral mucocele develops following traumatic severance of minor salivary gland duct and resultant spillage of mucous secretion into the surrounding connective tissues.[1],[2],[3],[4],[5],[6],[7] Its clinical presentation is very often distinctive as raised dome-shaped, circumscribed, nontender, sessile, and fluctuant swelling varying in size. Oral mucoceles are not true cysts and are distinct from the well-delineated, epithelium-lined mucous retention cyst or salivary duct cyst which usually develops from proximal expansion of a blocked duct.[1],[2]

Congenital mucous extravasation phenomenon is rare, and only sporadic reports have been reported.[3],[4],[5],[6],[7],[8],[9],[10],[11],[12],[13],[14] We could trace only one published case report for bilateral congenital mucous extravasation phenomenon.[9] The purpose of this paper is to discuss a rare case of bilateral congenital oral mucocele on buccal mucosa near commissure of mouth in an infant.


   Case Report Top


A 7-month-old male child reported with bilateral fluid-filled nodules over labial mucosa near the commissure of mouth. The nodules presented as smooth, sessile, normal-colored, and fluctuant of size approximately measuring 4 mm × 4 mm [Figure 1]. The lesions were noticed at birth, subsequently enlarged to interfere with feeding. The lesion frequently ruptured, drained, and again enlarged to interfere with normal function. Parents reported a history of otherwise healthy and normally delivered baby. The history, clinical presentation, and differential diagnosis suggested a provisional diagnosis of congenital mucous extravasation phenomenon or bilateral oral mucocele. Although the lesion was identified at birth by the parents, treatment was carried out 11 months after birth which was due to absence of symptoms in between phases due to frequent rupture and drain of the mucocele repeatedly.
Figure 1: Congenital bilateral oral mucocele on buccal mucosa near the commissure of mouth

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The lesions were observed for progressive enlargement or regression over a period of 4 months and then the decision was made to excise both the lesions. Parental consent and preanesthetic clearance were obtained. Both lesions were surgically excised under general anesthesia [Figure 2]a and [Figure 2]b at the age of 11 months using electrocautery after local infiltration of 2% lignocaine with 1:100000 adrenaline solution. The child recovered normal feeding after 24 h without any postoperative complication. The histopathological examination of both specimens confirmed the diagnosis of mucocele [Figure 3]a,[Figure 3]b,[Figure 3]c. Postoperative evaluation showed normal healing, and follow-up after 3 months [Figure 4] showed no signs of recurrence. The patient was followed up telephonically at 6 months and 14 months, which revealed no recurrence of lesion.
Figure 2: (a) Superficiality of the lesion and excision with electrosurgery unit. (b) Intraoral view immediately after excision of lesion bilaterally

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Figure 3: (a) Excised lesion – specimen for histopathological examination. (b) Photomicrograph of lesion (H and E, ×100) showing mucosal epithelium and granulation tissue with collected mucin and inflammatory cells. (c) Photomicrograph of lesion (H and E, ×200)

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Figure 4: Follow-up intraoral view at 3 months after surgery

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   Discussion Top


The mucous extravasation phenomenon occurs most commonly in children in the second decade of life, with an incidence of generally 2.5 lesions per 1000 patients.[2],[14],[15] The exact incidence of oral mucocele is not known because spontaneously resolved [5],[12],[14] cases go unreported. Mínguez-Martinez et al. reported that 44% of mucoceles resolved spontaneously, of which several patients were infants.[14] Similarly, in a recent literature review by Shapira et al., it was reported that 19% of oral mucocele cases were known to resolve spontaneously.[5] However, congenital or neonatal occurrence is rare with sporadic reports only.[3],[4],[5],[6],[7],[8],[9],[10],[11],[12],[13],[14] Jorgenson et al.[16] in their study reporting oral lesions from 2164 healthy neonates noted 15 neonates with raised, fluid-filled lesions of unknown diagnosis as well as a congenital ranula. The descriptions are clinically suggestive of MEC and might represent a 0.74% incidence. Finkelstein et al. in a literature review of 1460 oral mucoceles found only 18 cases in infants, of which only two cases were identified as congenital mucocele.[4] According to Catoldo and Mosadami who reviewed a series of 594 cases of mucoceles, only 2.7% of their cases with mucocele were found in age < 1 year.[7] In another retrospective analysis of 89 cases of mucocele in children, Mínguez-Martinez et al. found 11 cases in infants and only four cases were congenital.[14] Shapira et al. found 7.1% incidence (n = 60) for mucocele as well as ranula cases in infants and neonates.[5] When their tabled data are analyzed for “mucocele in neonates” only (n = 11), the incidence comes to be 1.3%. The bilateral occurrence of oral mucocele is so rare that only one case has been known to us at present.[9] Porter et al.[17] reported a case of multiple salivary mucoceles but that was in a 4-year-old young boy. The PubMed search of English literature with key words such as “congenital mucous extravasation cyst,” “congenital labial mucocele,” “congenital bilateral mucocele oral,” and “congenital mucocele oral” showed 9, 12, 2, and 22 search results, respectively. On detailed analysis and cross-referencing of these search results, we could identify only one case of bilateral congenital oral mucocele.

The common cause of MEC is postulated to be trauma to minor salivary gland. The repeated trauma with pacifier to relieve teething irritation [10] or accidental biting of lower lip/tongue [8] has been postulated to be the possible etiology of oral mucocele in infants. In the present case, the basis for rare congenital presentation is not clear; however, in utero oral mucosal injury due to in utero thumb-sucking or trauma while passage through the delivery canal/forceps use or congenital atresia of minor salivary gland duct or manipulation of the neonate during the initial hours of birth may be the possible etiological factors.[5],[7],[18],[19] Some authors believe that oral mucocele may develop initially as mucous retention phenomenon due to excessively viscous saliva [12] which subsequently ruptures due to lower resistance of ductal epithelium in neonates/infants,[13] thus characterizing an extravasation phenomenon.[8]

Mucoceles are mainly diagnosed by clinical examination and with a history of previous trauma. However, in case of congenital lesion, oral mucocele should be differentiated from exophytic lesions, namely, eruption cysts, congenital epulis, melanotic neuroectodermal tumor of infancy, lymphangioma, hemangioma, lipoma, venous varix, salivary gland nodule/neoplasm, soft-tissue abscess, and cysticercosis.[1],[2],[5],[7],[9] The eruption cysts are not found on lip or buccal mucosa and were thus eliminated from differential diagnosis for the present case.[1] The differentiating features of congenital epulis from mucocele are: (1) smooth-surfaced soft mass and not cystic in consistency, (2) usually stop enlarging after birth and never repetitively regress and recur, (3) the most common site of occurrence is alveolar ridge, and (4) comprises large granular cells.[9],[20] The melanotic neuroectodermal tumor of infancy is differentiated from mucocele in: (1) its dark color, (2) locally destructive nature, (3) being the common site in anterior maxilla, and (4) histological biphasic larger melanin cells and smaller neuroblastic-appearing cells.[1],[9] Congenital oral hemangioma can also present as raised red/blue lesions, but unlike mucocele, the hemangioma blanches with diascopy.[21] Congenital oral lymphangioma can also present superficially as exophytic pebbled clear vesicular lesions.[1],[21] The appearance of mucocele is pathognomonic generally, but additional investigations such as fine-needle aspiration are useful diagnostic tools for evaluating salivary gland nodule and angiomatous lesion.[13] In some cases, routine radiographs, ultrasonography, computed tomography, or magnetic resonance imaging may be required to establish the extent and provisional diagnosis of lesion.[13] The histopathological examination is crucial to confirm the final diagnosis of mucocele.

Shapira and Akrish [5] reported that 85% of mucocele and ranula cases were surgically excised, whereas only 8% and 6% of cases were treated with marsupialization and decompression, respectively. In the present case, the surgical excision was performed as the lesion did not resolve spontaneously after wait and watch for 4 months till the age of 11 months. Though the surgical excision is the most common treatment modality, electrosurgery, cryosurgery, micro-marsupialization, and laser surgery are also proven to be equally effective.[3],[20],[22],[23],[24] The different techniques offer their own advantages and shortcomings. Wu et al.[25] used carbon dioxide laser vaporization in thirty patients younger than 18 years, of which only six cases were aged 0–6 years and compared them with 34 children with surgical excision and found no statistically significant difference between the two methods. Other known treatment methods such as cryosurgery and corticosteroid injections were not documented in neonates and infants. We were able to find only one study by Moraes Pde et al.[23] where they used liquid nitrogen cryosurgery in two children. In the present case, surgical excision with electrosurgery was opted considering the: (a) ease of performance because of superficiality, (b) small base, (c) clear demarcation with healthy tissue, and (d) eliminating the need for sutures and nonavailability of lasers. The excision with electrosurgery required <5 min to complete the procedure and excellent healing was reported without any recurrence. Commercial advertisements have stimulated the use of dental lasers by expressing their advantages. A comparison of diode laser with electrosurgery reveals significant overlap of potential uses and effectiveness. The decision regarding which modality to use is up to the individual doctor.[22]


   Conclusion Top


This study reported and discussed the second case of bilateral congenital mucocele. The clinical diagnosis, differential diagnosis, and management of such lesions in an infant may be challenging for a pediatric dentist. However, the superficial mucocele can be successfully treated with simple means without any complications.

Financial support and sponsorship

Nil.

Conflicts of interest

There are no conflicts of interest.



 
   References Top

1.
Neville BW, Damm DD, Allen CM, Bouquot JE. Oral and Maxillofacial Pathology. 1st ed. Philadelphia: W.B. Saunders Co.; 1995. p. 322-6, 390-7.  Back to cited text no. 1
    
2.
More CB, Bhavsar K, Varma S, Tailor M. Oral mucocele: A clinical and histopathological study. J Oral Maxillofac Pathol 2014;18 Suppl 1:S72-7.  Back to cited text no. 2
    
3.
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. 3
    
4.
Finkelstein MW, Hammond HL, Ebetino F Jr. Congenital mucous retention phenomena. ASDC J Dent Child 1984;51:379-81.  Back to cited text no. 4
    
5.
Shapira M, Akrish S. Mucoceles of the oral cavity in neonates and infants – Report of a case and literature review. Pediatr Dermatol 2014;31:e55-8.  Back to cited text no. 5
    
6.
Crean SJ, Connor C. Congenital mucoceles: Report of two cases. Int J Paediatr Dent 1996;6:271-5.  Back to cited text no. 6
    
7.
Gatti AF, Moreti MM, Cardoso SV, Loyola AM. Mucus extravasation phenomenon in newborn babies: Report of two cases. Int J Paediatr Dent 2001;11:74-7.  Back to cited text no. 7
    
8.
Cajazeira MR, Marins MR, Abrahão CA, Primo LG. Mucus Extravasation phenomenon in a newborn: An uncommon case. J Dent Child 2014;81:166-70.  Back to cited text no. 8
    
9.
Peters E, Kola H, Doyle-Chan W. Bilateral congenital oral mucous extravasation cysts. Pediatr Dent 1999;21:285-8.  Back to cited text no. 9
    
10.
Alves LA, Di Nicoló R, Ramos CJ, Shintome L, Barbosa CS. Retention mucocele on the lower lip associated with inadequate use of pacifier. Dermatol Online J 2010;16:9.  Back to cited text no. 10
    
11.
Kalra N, Chaudhary S, Singh B. Mucus extravasation phenomenon on the alveolar ridge in neonate: A case report. J Indian Soc Pedod Prev Dent 2004;22:36-7.  Back to cited text no. 11
[PUBMED]    
12.
Cunha RF, De M, Carvalho P, Guimaraes CM, Macedo CM. Surgical treatment of mucocele in an 11 month-old baby: A case report. J Clin Pediatr Dent 2002;26:203-6.  Back to cited text no. 12
    
13.
Kaneko T, Horie N, Shimoyama T. Congenital mucocele in the tongue: Report of a case. J Oral Maxillofac Surg 2012;70:2596-9.  Back to cited text no. 13
    
14.
Mínguez-Martinez I, Bonet-Coloma C, Ata-Ali-Mahmud J, Carrillo-García C, Peñarrocha-Diago M, Peñarrocha-Diago M, et al. Clinical characteristics, treatment, and evolution of 89 mucoceles in children. J Oral Maxillofac Surg 2010;68:2468-71.  Back to cited text no. 14
    
15.
Ata-Ali J, Carrillo C. Oral mucocele: Review of the literature. J Clin Exp Dent 2010;2:10-3.  Back to cited text no. 15
    
16.
Jorgenson RJ, Shapiro SD, Salinas CF, Levin LS. Intraoral findings and anomalies in neonates. Pediatrics 1982;69:577-82.  Back to cited text no. 16
    
17.
Porter SR, Scully C, Kainth B, Ward-Booth P. Multiple salivary mucoceles in a young boy. Int J Paediatr Dent 1998;8:149-51.  Back to cited text no. 17
    
18.
Granholm C, Olsson Bergland K, Walhjalt H, Magnusson B. Oral mucoceles; extravasation cysts and retention cysts. A study of 298 cases. Swed Dent J 2009;33:125-30.  Back to cited text no. 18
    
19.
Yagüe-García J, España-Tost AJ, Berini-Aytés L, Gay-Escoda C. Treatment of oral mucocele-scalpel versus CO2 laser. Med Oral Patol Oral Cir Bucal 2009;14:e469-74.  Back to cited text no. 19
    
20.
Damm DD, Cibull ML, Geissler RH, Neville BW, Bowden CM, Lehmann JE, et al. Investigation into the histogenesis of congenital epulis of the newborn. Oral Surg Oral Med Oral Pathol 1993;76:205-12.  Back to cited text no. 20
    
21.
Silverman RA. Hemangiomas and vascular malformations. Pediatr Clin North Am 1991;38:811-34.  Back to cited text no. 21
    
22.
Christensen GJ. Soft-tissue cutting with laser versus electrosurgery. J Am Dent Assoc 2008;139:981-4.  Back to cited text no. 22
    
23.
Moraes Pde C, Teixeira RG, Thomaz LA, Arsati F, Junqueira JL, Oliveira LB, et al. Liquid nitrogen cryosurgery for treatment of mucoceles in children. Pediatr Dent 2012;34:159-61.  Back to cited text no. 23
    
24.
Piazzetta CM, Torres-Pereira C, Amenábar JM. Micro-marsupialization as an alternative treatment for mucocele in pediatric dentistry. Int J Paediatr Dent 2012;22:318-23.  Back to cited text no. 24
    
25.
Wu CW, Kao YH, Chen CM, Hsu HJ, Chen CM, Huang IY,et al. Mucoceles of the oral cavity in pediatric patients. Kaohsiung J Med Sci 2011;27:276-9.  Back to cited text no. 25
    


    Figures

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



 

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