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신생아 급성 기도폐색을 유발한 후두개곡 낭종 2예

Vallecular Cyst Inducing Acute Airway Obstruction in Neonate : Two Cases

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주제분류이비인후과학

초록

Vallecular cyst is a rare in children. Vallecular cyst consists of a unilocular cystic mass of variable size arising from the lingual surface of the epiglottis and containing clear, non-infected fluid. Vallecular cyst has been reported in the literature under different names and this has led to some confusion. Terms used have included mucous retention cyst, epiglottic cyst, base-of-the-tongue cyst, congenital cyst and more recently ductal cyst. This later name originates from the classification of DeSanto et al, in which they grouped laryngeal cysts according to their location and surface mucosa (Thyroid cartilage foraminal cysts ; Saccular cysts ; Ductal cysts). This classification has become popular but has the limitation that, it was largely based on observation in adults and did not recognize different anatomical sites and variations in clinical presentation. Although these cysts are benign, they may cause serious airway obstruction and even death if not treated appropriately. A mortality rate of about 40% in children with laryngeal cysts was reported1. Vallecular cyst or pre-epiglottic cyst is a rare but recognized cause of respiratory distress in infancy or immediately after birth. It has been associated with sudden airway obstruction resulting in death. We experienced two cases of vallecular cyst with feeding difficulty and stridor in 2-month old female and dyspnea in 4-month old male who symptom relief by endoscopic laser-assisted resection. With a review of literature, authors report two cases of vallecular cyst with the symptoms of upper airway compromise.

Abstract

Vallecular cyst is a rare in children. Vallecular cyst consists of a unilocular cystic mass of variable size arising from the lingual surface of the epiglottis and containing clear, non-infected fluid. Vallecular cyst has been reported in the literature under different names and this has led to some confusion. Terms used have included mucous retention cyst, epiglottic cyst, base-of-the-tongue cyst, congenital cyst and more recently ductal cyst. This later name originates from the classification of DeSanto et al, in which they grouped laryngeal cysts according to their location and surface mucosa (Thyroid cartilage foraminal cysts ; Saccular cysts ; Ductal cysts). This classification has become popular but has the limitation that, it was largely based on observation in adults and did not recognize different anatomical sites and variations in clinical presentation. Although these cysts are benign, they may cause serious airway obstruction and even death if not treated appropriately. A mortality rate of about 40% in children with laryngeal cysts was reported1. Vallecular cyst or pre-epiglottic cyst is a rare but recognized cause of respiratory distress in infancy or immediately after birth. It has been associated with sudden airway obstruction resulting in death. We experienced two cases of vallecular cyst with feeding difficulty and stridor in 2-month old female and dyspnea in 4-month old male who symptom relief by endoscopic laser-assisted resection. With a review of literature, authors report two cases of vallecular cyst with the symptoms of upper airway compromise.

참고문헌 (13)

  1. Wong KS, "Vallecular cyst synchronous with laryngomalacia: presentation of two cases", Otolaryngol Head Neck Surg, 113(5), pp.621~624 (1995)
  2. Ruben RJ, "Cystic lymphangioma of the vallecula", Can J Otol, 4(1), pp.180~184 (1975)
  3. Gutierrez JP, "Vallecular cysts in newborns and young infants", Pediatr Pulmonol, 27(4), pp.282~285 (1999)
  4. Wong KS, "A vanishing tongue-base cyst", Turk J Pediat, 49(4), pp.451~452 (2007)
  5. DeSanto LW, "Cysts of the larynx classification", Laryngoscope, 80(1), pp.145~176 (1970)
  6. Yao TC, "Failure to thrive caused by the coexistence of vallecular cyst, laryngomalacia and gastroesophageal reflux in an infant", Int J Pediatr Otorhinolaryngol, 68(11), pp.1459~1464 (2004)
  7. Tibesar RJ, "Apnea spells in an infant with val-lecular cyst", Ann Otol Rhinol Laryngol, 112(9), pp.821~824 (2003)
  8. Garel C, "Laryngeal ultrasonography in infants and children: anatomical correlation with fetal preparations", Pediatr Radiol, 20(4), pp.241~244 (1990)
  9. Garel C, "Laryngeal ultrasonography in infants and children: pathological findings", Pediatr Radiol, 21(3), pp.164~167 (1991)
  10. Imhof H, "Tumors and tumor-like lesions of the neck: from childhood to adult", Eur Radiol, 14 Suppl, pp.L155~L165 (2004)
  11. Tuncer U, "Vallecular cyst: a cause of failure to thrive in an infant", Int J Pediatr Otorhinolaryngol, 65(2), pp.133~135 (2002)
  12. Koch BL, "Cystic malformations of the neck in children", Pediatr Radiol, 35(5), pp.463~477 (2005)
  13. Ku AS, "Vallecular cyst: report of four cases-one with co-existing laryngomalacia", J Laryngol Otol, 114(3), pp.224~226 (2000)

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