Korean J Perinatol.  2014 Mar;25(1):22-26. 10.14734/kjp.2014.25.1.22.

A Case of Intrauterine Lingual Cyst with Successful Delivery Assisting Ex Utero Intrapartum Treatment Procedure

  • 1Department of Pathology, Keimyung University School of Medicine, Daegu, Korea.
  • 2Department of Obstetrics and Gynecology, Keimyung University School of Medicine, Daegu, Korea. gonmd@dsmc.or.kr


Congenital lingual cyst is rare and there has been no prior case report about prenatally detected lingual cyst in Korea. When a huge oral cyst is observed at prenatal period and can cause life-threatening airway obstruction at birth, ex utero intrapartum treatment (EXIT) procedure is needed to secure the airway. Herein we present a baby with a prenatally detected huge oral cyst. He was delivered safely assisting EXIT procedure and underwent an operation for resection of the cyst from his tongue. The oral cyst was diagnosed as a lingual cyst with rare histologic type consisting mixed gastrointestinal and respiratory epithelium.


Lingual cyst; Ex utero intrapartum treatment procedure; Congenital cyst; Oral cavity; Tongue

MeSH Terms

Airway Obstruction
Respiratory Mucosa


  • Fig. 2. A large lingual cyst with risk of respiratory obstruction is present in oral cavity.

  • Fig. 1. Ultrasonography at 26 weeks of gestational age shows a large cystic lesion in oral cavity of fetus (A). MRI at 32 weeks of gestational age also shows a large simple cystic mass (34×31×27 mm, arrow) without solid portion or septal formation (B).

  • Fig. 3. The cyst is consisted mixed gastrointestinal and respiratory epithelium. Cystic lining cells are included gastrointestinal cells such as mucus, parietal or chief cells (A, HE stain, × 200). Cystic lining cells are also included ciliated pseudostratified columnar cells (respiratory epithelium) and squamous cells (B, HE stain, ×200).


1.Jing SS., Alberry MS., Smith RP., Clibbon JJ. A rare report of foetal lingual cyst excised with harmonic scalpel. J Plast Re-constr Aesthet Surg. 2013. 66:1770–2.
2.Azanero WD., Mazzonetto R., Leon JE., Vargas PA., Lopes MA., de Almeida OP. Lingual cyst with respiratory epithelium: a histopathological and immunohistochemical analysis of two cases. Int J Oral Maxillofac Surg. 2009. 38:388–92.
3.Manor Y., Buchner A., Peleg M., Taicher S. Lingual cyst with respiratory epithelium: an entity of debatable histogenesis. J Oral Maxillofac Surg. 1999. 57:124–7. discussion 8-9.
4.Mychaliska GB., Bealer JF., Graf JL., Rosen MA., Adzick NS., Harrison MR. Operating on placental support: the ex utero intrapartum treatment procedure. J Pediatr Surg. 1997. 32:227–30. discussion 30-1.
5.Skarsgard ED., Chitkara U., Krane EJ., Riley ET., Halamek LP., Dedo HH. The OOPS procedure (operation on placental support): in utero airway management of the fetus with prenatally diagnosed tracheal obstruction. J Pediatr Surg. 1996. 31:826–8.
6.Ngamprasertwong P., Vinks AA., Boat A. Update in fetal anesthesia for the ex utero intrapartum treatment (EXIT) procedure. Int Anesthesiol Clin. 2012. 50:26–40.
7.DeCou JM., Jones DC., Jacobs HD., Touloukian RJ. Successful ex utero intrapartum treatment (EXIT) procedure for congenital high airway obstruction syndrome (CHAOS) owing to laryngeal atresia. J Pediatr Surg. 1998. 33:1563–5.
8.Dighe MK., Peterson SE., Dubinsky TJ., Perkins J., Cheng E. EXIT procedure: technique and indications with prenatal imaging parameters for assessment of airway patency. Radio-graphics. 2011. 31:511–26.
9.Joshi R., Cobb AR., Wilson P., Bailey BM. Lingual cyst lined by respiratory and gastric epithelium in a neonate. Br J Oral Maxillofac Surg. 2013. 51:173–5.
10.Bite U., Cramer HM. Mixed heterotopic gastrointestinal and respiratory cyst of the lip: case report and review of the literature. Plast Reconstr Surg. 1992. 90:1068–72.
11.Burkart CM., Brinkman JA., Willging JP., Elluru RG. Lingual cyst lined by squamous epithelium. Int J Pediatr Otorhino-laryngol. 2005. 69:1649–53.
12.Ohbayashi Y., Miyake M., Nagahata S. Gastrointestinal cyst of the tongue: a possible duplication cyst of foregut origin. J Oral Maxillofac Surg. 1997. 55:626–8. discussion 9-30.
13.Edwards RM., Chapman T., Horn DL., Paladin AM., Iyer RS. Imaging of pediatric floor of mouth lesions. Pediatr Radiol. 2013. 43:523–35.
14.Bae JG., Park JC., Kwon SH., Cho CH., Rhee JH., Cha SD. A case of cervical branchial cleft cyst diagnosed with antenatal ultrasonography in fetus. Korean J Obstet Gynecol. 2008. 51:900–4.
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