A male newborn who weighed 3480 g was delivered vaginally, with Apgar scores of 10 and 10 at 1 and 5 minutes, respectively. The neonate had no feeding or breathing difficulty. No major oral cyst was identified in the tongue at a clinical and instrumental examination. We followed the child to 6 years after birth. The child had normal growth and development, with no recurrence of the cyst. The study was approved by the Institute Research Ethic Committee of Peking Union Medical College Hospital. The patient has provided informed consent for publication of the case.
A search of PubMed showed only 18 cases of lingual cysts that were diagnosed prenatally. These cases included six ranulas, seven foregut duplication cysts, 2 thyroidglossal duct cysts, one lymphangioma, and one cystic epignathus. Prenatal recognition of such a tumor should prompt a thorough evaluation of the likelihood of airway tract and esophageal obstruction. CDFI and magnetic resonance imaging (MRI) provide the ability to visualize a fluid-filled oral cavity, oropharynx, nasopharynx, and trachea. Real-time ultrasound and MRI can demonstrate motion of the mass during swallowing. Normal amniotic fluid volume and visualization of stomach bubbles are helpful for determining if there is no distortion or compression of the esophagus. Routine follow-up should be scheduled bi-weekly to monitor growth of the lesion. Steelman et al reported a ranula that spontaneously resolved at 5 weeks of age. However, a cystic lesion that disappeared before birth has not been reported. In some of the reported cases, a considerable increase in lesion size required an intervention to relieve obstruction.[4,9,15] Pires et al and George conducted ultrasound-guided aspiration of the cyst at 26 weeks of gestation; Deloison performed cyst puncture immediately prior to delivery. Prenatal decompression of the cyst can prevent postnatal airway obstruction, especially if carried out just before birth, which can also avoid the risk of hemorrhage from the cyst. Two of the reviewed cases underwent an extrauterine intrapartum treatment procedure with aspiration of the cyst to secure the airway of the infant.[16,17] This technique allows maintenance of uteroplacental gas exchange in the newborn. The definitive treatment for these tumors is postnatal surgical resection.
We report a case of a relatively small cyst with spontaneous in utero resolution. A lingual cyst usually has a good prognosis. When a lingual cyst is prenatally diagnosed, the patency of the airway tract and esophagus should be evaluated. Prenatal aspiration of the cyst or the extrauterine intrapartum treatment procedure facilitates a successful outcome for large lingual cysts.
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