Intra-oral enteric duplication cysts are rare. We recently treated two infants with prenatally diagnosed large sublingual enteric duplication cysts. Antenatal aspiration of the cyst was attempted in one infant without success. Because of the potential for airway obstruction and respiratory distress at delivery, preparation for an immediate tracheostomy was made in case oral intubation was impossible. Fortunately, aspiration of the cyst provided adequate access to the oropharynx for intubation in both infants. The placental cord was not divided until the airway was secured to minimize the potential for anoxic injury. Complete excision of the cyst was subsequently done in one patient. The other infant had an initial marsupialization and laser ablation of the cyst mucosa but the cyst recurred. Cyst excision at 9 months of age resolved the problem. Pathological examination revealed columnar-lined mucosa consistent with enteric duplication cysts. An enteric duplication cyst may present as a large sublingual mass with potential for airway obstruction in the newborn infant. Antenatal diagnosis allows for the proper preparation of personnel and equipment in the management of these neonates during delivery. If the airway is compromised, aspiration of the cyst can be done to improve the access to the oropharynx for intubation. Preparation for emergent tracheostomy should be done before delivery in case oral intubation is not possible. Subsequent complete cyst excision is the treatment of choice.