Congenital oral cysts in neonates: report of two casesAnup Mohta, MS, MCh,a and Mrigank Sharma, MS,b Delhi, IndiaUNIVERSITY COLLEGE OF MEDICAL SCIENCES AND GURU TEG BAHADUR HOSPITAL
Oral cysts in the neonatal period are very uncommon and may cause morbidity and mortality if not treatedexpeditiously. We report 2 cases of neonatal oral cysts that were managed successfully. (Oral Surg Oral Med Oral
Pathol Oral Radiol Endod 2006;102:e36-e38)
Pediatric surgeons, pediatricians and oral-maxillofacialsurgeons are faced with a spectrum of oral cystic le-sions. These are uncommon in neonates, and if notrecognized and treated expeditiously, these anomaliesmay obstruct the upper aero-digestive tract and be fatal.We report 2 successfully managed cases of congenitaloral cysts in neonates that presented with masses in theoral cavity.
CASE REPORTSCase 1
A 2-day-old male neonate presented to hospital with alarge mass in the oral cavity and complaints of inability tofeed and respiratory difficulty. A large tense nontransillumi-nant, intralingual lesion was seen obliterating the oral cavity(Fig. 1). Aspiration of about 20 mL of brownish fluid from thecyst decompressed the swelling, confirmed its intralingualposition and relieved the respiratory difficulty. A nasogastrictube was inserted and intravenous fluids and antibiotics wereadministered. As the cyst refilled overnight along with per-sistent difficulty in respiration and feeding, emergency sur-gery was planned.
After preoperative aspiration of the cyst to facilitate intu-bation, the intralingual cyst was completely dissected bysagittal incision. After securing hemostasis, the tongue wasreconstructed. The child was extubated after surgery whenfully awake but equipment for intubation was kept ready incase of any need. The patient was fed expressed mother’smilk through the nasogastric tube beginning 48 hours aftersurgery. Postoperative edema of the tongue resolved in next 7days. Histopathology showed mucus-secreting columnar ep-ithelium and focal areas of squamous lining suggestive ofenterogenous duplication cyst. The patient is well at 6 monthsfollow-up.
Case 2A 15-day-old female child presented to the hospital with a
swelling in the floor of the mouth since birth (Fig. 2). Parentsgave a history of difficulty in feeding since birth. The childwas febrile with tachypnea and was not accepting food. A 4� 4-cm size tense cystic lesion was seen in the floor of themouth that had pushed the tongue toward the palate. Aclinical impression of infected ranula in the floor of the mouthwas made and the patient was taken up for emergency sur-gery. In view of the anticipated difficult intubation, preoper-ative aspiration of 7 mL of yellowish thick fluid was done.Marsupialization of the cyst was performed as an emergencyprocedure and child was administered antibiotics. Oral feed-ing was started on the third postoperative day. Histopathologyof the excised cyst wall was suggestive of epidermoid cyst.Recovery was uneventful and the child was discharged after7 days and was well without recurrence at 1-year follow-up.
DISCUSSIONOccurrence of cystic lesions in the oral cavity is
Fig 1. Photograph showing large intralingual enterogenousduplication cyst.
uncommon in neonates. Various causes of intraoral
pushin
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cystic lesions include enteric duplication cysts, dermoidand epidermoid cyst, hemangioma, ranula, lingual thy-roid, lymphoepithelial cyst, cystic hygroma, and tera-toma.1 If large in size these can cause difficulty infeeding because of oropharyngeal obstruction and oc-casionally respiratory difficulty because of airway ob-struction.
Oral cavity duplication cysts constitute a small num-ber with about 0.3% present in the tongue1 and intra-lingual enterogenous cysts are even more uncommon.Theories of pathogenesis of enteric duplications includedevelopment from small epithelial inclusions trappedduring fusion of primordial tissues, from incompletecoalescence of lacunas that form between epithelialcells of the solid core of the developing gut, frompersistence of epithelial buds within the wall of thebowel, or from nests of trapped entodermal cells.2
The presentation is usually in the first decade of lifeand is very rare in the neonatal age group. There is apredilection for males. Presentation depends on sizeand location, and may cause airway compromise anddysphagia. Complete excision by sagittal lingual split ispossible and preferred in most cases of intralingualcysts of foregut origin.3 Aspiration alone is inadequateand may lead to secondary infection, whereas marsu-pialization may lead to recurrence. Presence of func-tional mucosa leads to mucus secretion and occasion-ally hemorrhage from heterotropic gastric mucosa ifleft intact.4
Dermoid and epidermoid cysts are developmentalanomalies that occur in the head and neck with anincidence ranging from 1.6% to 6.9%, and they accountfor less than 0.01% of all oral cavity cysts.5 They
usually present as a nonpainful swelling. Dermoid cysts
of the floor of the mouth are thought to be caused byentrapment of germinal epithelium during the closureof the mandibular and hyoid branchial arches. Histo-logically, all dermoids are lined by epidermis. Thecontents of the cyst lining determine the histologicalcategories of the cyst: epidermoid, if epidermis is liningthe cyst; dermoid, if skin adenxa exist; or teratoid, ifthere are tissues derived from the 3 germinal layers.6
Congenital lingual cystic masses are challenging en-tities that can be detected prenatally using ultrasound. Ifa lesion is detected in the fetal mouth or neck, furtherassessment and monitoring can be done using fetalmagnetic resonance imaging (MRI).7
Antenatal diagnosis allows for proper preparation ofpersonnel and equipment for management in postnatalmanagement. Antenatal aspiration is difficult. The pla-cental cord should be left undivided until the airway issecured.4 Although rarely required, preparation foremergency tracheostomy should be kept ready. Theex-utero intrapartum (EXIT) procedure has recentlybeen developed to allow lifesaving fetal surgery to beperformed during delivery of such cases while relyingon placental support.7
The preoperative evaluation of lingual cystic massesin newborns includes palpation, high-resolution sonog-raphy, computed tomography (CT), or MRI. However,CT and MRI require sedation in order to properlyposition the neonate and can pose a risk. High-resolu-tion sonography has been found to be very useful inrevealing the nature of the mass and in delineating itsextension.8
At anesthetic induction, aspiration allows visualiza-tion of vocal cords and subsequent tracheal intubation.
g the tongue toward the roof of the oral cavity.
Both cases presented underwent preoperative aspiration
OOOOEe38 Mohta and Sharma November 2006
to facilitate intubation. Smaller cysts can be com-pressed into the submandibular region to facilitate la-ryngoscopy and intubation.
Postoperative airway and respiratory managementneeds to be closely monitored and a nasopharyngealairway should be available. Postoperative swelling ofthe tongue can occur, which can require intubation attimes. No airway difficulty was experienced by eitherof these patients in the postoperative period. Earlyfeeding with mother’s milk may be resumed after sur-gery through the nasogastric tube as was done in one ofour patients.
Successful management of the presented cases em-phasizes the need of early diagnosis and prompt surgi-cal intervention for optimal management of neonataloral cysts.
REFERENCES1. Tucker R, Maddalozzo J, Choiu P. Sublingual enteric duplication
cyst. Arch Pathol Lab Med 2000;124:614-5.
2. Lipsett J, Sparnon AL, Byard RW. Embryogenesis of enterocys-tomas-enteric duplication cysts of the tongue. Oral Surg Oral MedOral Pathol 1993;75:626-30.
3. Wiersma R, Hadley GP, Bosenberg AT, Chrystal V. Intralingualcysts of foregut origin. J Pediatr Surg 1992; 27:1404-6.
4. Chen MK, Gross E, Lobe TE. Perinatal management of entericduplication cysts of the tongue. Am J Perinatol 1997;14:161-3.
5. De Ponte FS, Brunelli A, Marchetti E, Bottini DJ. Sublingualepidermoid cyst. J Craniofac Surg 2002;13:308-10.
7. Hall NJ, Ade-Ajayi N, Peebles D, Pierro A. Antenatally diagnosedduplication cyst of the tongue: modern imaging modalities assistperinatal management. Pediatr Surg Int 2005;21:289-91.
8. el-Bitar MA, Milmoe G, Kumar S. Intralingual foregut duplicationcyst in a newborn. Ear Nose Throat J 2003;82:454-6.
