Congenital Mucocele of the Tongue: A case report with review of the literature

INTRODUCTION

Congenital Mucocele of tongue is a very uncommon condition where the infant presents with airway obstruction, feeding difficulty, respiratory distress and failure to thrive. Prenatal detection of these lesions is important since they may cause breathing problems at birth and interfere with breastfeeding¹ .

An approximate annual incidence of congenital Mucocele is 1.82 per 100000 live births in an oriental population was reported¹ . The incidence of retention cyst in oral cavity is much more common in the general population and is estimated as 2.4 cases per 1,000 populations, frequently occurring in the second decade of life^2,3. Mucocele involving the ventral surface of the tongue are very rare, with sporadic cases being reported in the world literature^4,5 accounting to 2-8% of oral mucocele⁶ .

A tongue cyst could remain completely asymptomatic and persist till adolescence with subsequent regression of the symptoms. A suspected case of tongue cyst should be followed up with routine investigations along with imaging studies to come to a conclusive diagnosis and to know the extent of the cyst.

CASE REPORT

A 5-day male neonate was presented with a history of swelling on the ventral aspect of tongue (Fig. 1) since birth and difficulty in breast feeding. The neonate was not able to close the mouth since birth with continuous protrusion of the tongue. There was no history of respiratory distress or cyanosis at birth.

The child had a normal full-term vaginal delivery with immediate cry after birth. Antenatal history of mother was uneventful. Antenatal ultrasonography was normal.

Oral examination revealed 3.5 × 2.5 cm sized tense swelling along the ventral surface of the tongue causing persistent mouth opening. Swelling had a smooth surface, nontender, circumscribed, nonulcerated round mass with cystic consistency. Swelling had a bluish colour and brightly transilluminant. The dorsum of the tongue had a normal in texture. The base of tongue could not be visualized. On ultrasonography, the swelling showed hypoechoic lesion with normal Doppler flow without any vascularity (Fig. 2).

Neonate underwent en block excision of the cystic swelling, with removal of entire cyst wall. Jelly like fluid was noticed after cyst opening (Fig. 3). The defect was closed with interrupted sutures (Fig. 4). Postoperative period was uneventful.

Histopathological examination showed cystic lesion with islets of salivary glands in the wall suggestive of mucocele (Fig. 5).

DISCUSSION

Tongue cyst is a rather rare occurrence with very few cases reported in the literature. Majority of cases with a tongue cyst are asymptomatic and would regress as age progresses. There are two major hypotheses to explain the pathogenesis, which consider the cyst is a consequence of either ductal obstruction of the mucus glands or an embryological malformation⁷ .

Majority of the mucocele of the oral cavity occur in the lower lip⁵ . Occasionally they may be seen on the ventral surface of the tongue where they involve glands of Blandine -Nuhn^2,6. They are sub mucous glands located within the ventral part of the muscles of the tongue, and are anteriorly placed close to the midline with their ducts opening on the inferior surface. Mucocele usually follow trauma and are seen in younger age group. But in our case, it was of congenital variety of mucocele of the tongue in the new-born.

Congenital mucocele on the ventral aspect/lingual surface of the tongue in less than 1 year of age are very uncommon.

As an effort to collect data we performed a comprehensive search of the literature using Pub Med/Medline electronic database, all published congenital mucocele on the ventral aspect of the tongue were reviewed, and their age of presentation, type of lesion, chief complaints are presented in table no ^18-11.

According to collected data, only four cases have been published with age of presentation less than 1 year: 1 case had prenatal diagnosis at 39 weeks of gestation. 2 cases had chief complaints in the form of breastfeeding difficulty and respiratory distress, which was seen in our case. The fourth case reported was of 4 months old with breathing and feeding difficulty.

On histopathological examination all the published cases had mucous retention cyst type of entity^8-11. All cases exhibited swelling on the ventral aspect / lingual surface of tongue, which was the presentation in our case also.

CONCLUSION

Tongue mucocele should be considered in differential diagnosis in new-born with tongue swelling presenting with feeding difficulty, stridor, respiratory distress. Definitive therapy requires marsupialization or complete excision of the cyst wall.

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