CASE REPORT Pediatric Dermatology Vol. 31 No. 2 e55–e58, 2014

Mucoceles of the Oral Cavity in Neonates and Infants—Report of a Case and Literature Review Moshe Shapira, M.D.* and Sharon Akrish, D.D.S.† *Maccabi Health Services, Nave Sha’anan Clinic/Pediatrics, Haifa, Israel, †Department of Pathology and Oral and Maxillofacial Surgery, Rambam Health Care Campus, Technion Rappaport Faculty of Medicine and Research Institute, Haifa, Israel

Abstract: Oral mucoceles are common lesions that normally appear in the second decade of life. They are uncommon in neonates and infants. We present a case of a mucocele in a 10-month-old infant and review the literature regarding the epidemiology and treatment in this age group.

Mucoceles are one of the more common lesions found in the oral cavity, accounting for 11.6% to 24.5% of all pediatric oral biopsies (1–3). Mucoceles commonly affect children but are rarely found in neonates and infants (4). Clinically, mucoceles present as “dome-shaped” intramucosal lesions and are most frequently located on the lower lip. Other intraoral sites include the ventral tongue (involving the glands of Blandin-Nuhn), palate, buccal mucosa, upper lip, and retromolar region (5). Mucoceles occur through two mechanisms: extravasation and retention. The more common extravasation type of mucocele is due to the rupture of a minor salivary gland duct and spillage of mucin into the surrounding tissue that then becomes encapsulated by granulation tissue. If located on the floor of the mouth they are called ranulas, derived from the Latin word rana (frog) because they resemble the underbelly of a frog. Ranulas most commonly involve the major salivary glands, originating in the Wharton’s duct of the

submandibular gland and in the body or the ducts of Rivini of the sublingual gland. They have a prevalence of 0.2 cases per 1,000 persons (2,4). Ranulas tend to grow larger than other mucoceles and may interfere with swallowing, mastication, speech, and breathing. Microscopically, extravasation mucoceles appear as a pseudocystic space surrounded by granulation tissue. Mucin, neutrophils, and macrophages may be found within the space. The second subset is the mucous retention cyst, which is caused by ductal obstruction, resulting in dilatation and accumulation of mucin. The retention cyst type of mucocele is lined with ductal epithelium and is considered a true cyst (5,6). This is the most common type seen in adults. CASE REPORT A 10-month-old boy presented with an exophytic nodule of 2 weeks duration located on the right side of the lower lip. His past medical history was

Address correspondence to Moshe Shapira, M.D., Maccabi Health Services, Nave Sha’anan Clinic, 73 Hathicon Street, Haifa, Israel, or e-mail: [email protected]. The authors wish to acknowledge and thank Igor Shapiro for his technical assistance in preparing the manuscript. DOI: 10.1111/pde.12264

© 2013 Wiley Periodicals, Inc.

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unremarkable; his first tooth appeared at 4 months of age. The lesion on his lip did not interfere with sucking or feeding. There was no history of trauma or surgical procedure to the oral cavity. The mother stated that the lesion first appeared as a white dot that progressively increased in size. The mother noted that the baby had a habit of repeatedly hitting his pacifier with his palms to relieve teething pain and had had excessive drooling before and during the appearance of the lesion. Clinical examination revealed a translucent, fluid-filled swelling that was soft in consistency and measured 1.5 cm in size (Fig. 1). He was referred to maxillofacial surgery, where a clinical diagnosis of mucocele was made. After a period of watch and wait, the lesion persisted and slightly increased in size. Surgical excision was decided upon. Microscopic examination revealed a soft tissue lesion covered by hyperplastic stratified squamous epithelium. There was a space within the connective tissue lined by granulation tissue and filled with neutrophils, macrophages, and mucin. A minor salivary gland with some atrophy of the acini and a slightly dilated duct was found adjacent to the mucocele. The histologic diagnosis was mucocele (extravasation type) (Fig. 2). No postoperative complications or recurrence were noted during 2 years of follow-up.

Figure 2. Photomicrograh of a granulation tissue lined pseudocyst filled with mucin, macrophages, and neutrophils. (Hematoxylin and eosin stain, original magnification 1009).

22 studies appropriate for our review (6–26). In our study we segregated patients into two categories: neonates (0–1 month) and infants (1–12 months). We also grouped the mucoceles into two categories: mucoceles occurring on the floor of the mouth (ranulas) and those in all other regions of the oral cavity (mucoceles). The total cohort consisted of 840 patients, 60 (7.14%) of whom were infants or neonates (30 in each group). The sex of the patients was known in only 46 cases and showed an almost equal distribution (22 boys, 24 girls). Mucoceles were more commonly found in boys in the neonate population (58%, vs 39% in infants). The location of mucoceles was identified in 43 cases. Overall, 24 cases (56%) were located on the floor of the mouth and designated as ranulas, and 44% were mucoceles. The mucocele cases were located in various regions throughout the oral cavity: lower lip (n = 10, 23%), buccal mucosa (n = 4, 9%), tongue (n = 3, 7%), alveolar ridge (n = 1, 2%) and uvula (n = 1, 2%). Ranulas were more common in neonates than in the infant population and represented 65% of all cases. Only 36% of infant cases were ranulas (Table 1). Ranulas in the adult population are

DISCUSSION Oral cavity mucoceles are uncommon in infants (4). A PubMed search of the English-language literature for “mucoceles,” plus our presenting case, resulted in

Figure 1. Mucocele of the lower lip.

TABLE 1. Location of Neonatal and Infant Mucoceles

Neonates (n = 30) Infants (n = 30) Total (N = 60)*

Floor of the mouth (ranula)

Lower lip

Tongue

Uvula

Alveolar ridge

Buccal mucosa

Unspecified

19 5 24 (56)

6 4 10 (23)

2 1 3 (7)

1 0 1 (2)

1 0 1 (2)

0 4 4 (9)

1 16 17

Values are given as number or number (percent). *Total cohort for percent calculation was 43.

Shapira and Akrish: Mucoceles of the Oral Cavity in Neonates and Infants

far less common than mucoceles. In one review, most lesions in adults were found to be mucus extravasation phenomenon-type mucoceles (91%) and only 6% were ranulas (4). The cause of mucoceles is often attributed to trauma. As in our case, infants sometimes acquire habits to relieve the symptoms associated with tooth eruption, which can cause damage to a salivary duct in a minor salivary gland (27). In neonates, trauma to a salivary duct may be attributed to sucking fingers in utero, passage through the delivery canal, forceps use during delivery, or manipulation of the neonate after birth (6,10). In addition, a true defect in the development of the duct system, such as duct duplications (24) or congenital atresia of the sublingual or submandibular duct orifices with imperforate ducts (11,14,15), may prevent normal saliva outflow, leading to the development of a mucocele. Zhi et al (18) reported 11 cases of ranula in which none of the patients had a history of trauma, a fact that may indicate a congenital cause. In addition, Pownell et al (24) and Addante (28) described five newborns born with congenital anomalies of the submandibular duct. These patients subsequently developed a ranula-like lesion. The clinical differential diagnosis for swellings in the floor of the mouth and oral cavity in the neonate and infant population include dermoid cysts, epidermoid cysts, eruption cysts, hemangiomas, and lymphangiomas (2,4,6,17). Lypka et al (29) emphasized that a swelling located on the retromolar pad (posterior to the mandibular deciduous molar) should be biopsied to distinguish a mucocele from the less common yet more dangerous salivary gland tumor, a mucoepidermoid carcinoma. In most cases, treatment for a ranula is surgical excision including the salivary gland (15). Spontaneous resolution of a mucocele is possible, especially when small or superficially located. In our review, treatment was known in 48 cases. The vast majority (mucoceles and ranulas) were surgically excised (n = 41, 85%). The remainder were treated with marsupialization (n = 4, 8%) and decompression (n = 3, 6%); 11 cases (19%) were known to resolve spontaneously. In one case the treatment was not mentioned. Martinez et al (20) reported that 39 of 89 (44%) pediatric mucocele cases resolved spontaneously, of which several patients were infants. Zhi et al (18) reported 11 infant ranula cases in which marsupialization (2 cases), decompression (2 cases), or surgical excision including the sublingual gland (7 cases) was performed. Wu et al (3) used carbon dioxide laser vaporization in the treatment of oral mucoceles in 30 patients younger than 18 years old

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(6 cases ages 0–6 years) and compared them with 34 children after surgical excision. He found no statistically significant difference between the two methods regarding recurrence rates. The advantage of carbon dioxide laser vaporization was proposed to be less bleeding, no sutures, and shorter treatment time. Other known treatment modalities, such as cryosurgery (freezing) and corticosteroid injections, were not documented in neonates and infants (20). In summary, 7.1% of evaluated mucocele and ranula cases were found to occur in neonates and infants, with an almost equal sex distribution between them. Ranulas were more commonly found in neonates than infants. The most common location was the floor of the mouth, designated as ranula, followed by the lower lip (mucocele). Trauma, such as biting, that resulted in damage to the minor salivary gland duct was often noted. A developmental defect such as congenital atresia of a salivary duct or duplication is another possible cause of mucocele development, especially in neonates. The treatment of choice is surgical excision. For large ranulas, marsupialization may be employed. Other treatment modalities such as carbon dioxide laser vaporization have been introduced.

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Mucoceles of the oral cavity in neonates and infants--report of a case and literature review.

Oral mucoceles are common lesions that normally appear in the second decade of life. They are uncommon in neonates and infants. We present a case of a...
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