Subglottic ductal cysts associated with complete tracheal ring deformity: coexistence of two rare airway abnormalities with fatal outcome

• Published in: Pediatric and developmental pathology Vol. 7; no. 3; pp. 273 - 276
• Main Authors: Bokhari, Aqiba, Heller, Debra S, Fonseca, Carlos, Mirani, Neena
• Format: Journal Article
• Language: English
• Published: United States 01.05.2004
• Subjects: Cysts - complications; Cysts - pathology; Fatal Outcome; Glottis - pathology; Humans; Infant; Laryngeal Diseases - complications; Laryngeal Diseases - pathology; Male; Trachea - abnormalities; Tracheal Stenosis - complications; Tracheal Stenosis - pathology
• ISSN: 1093-5266; 1615-5742
• DOI: 10.1007/s10024-003-0106-5

Subglottic cysts are rare and may cause airway obstruction. Most cases are acquired secondary to endotracheal intubation, even short-term, particularly in the premature neonate. Complete tracheal rings are rare anomalies associated with tracheal stenosis. To our knowledge, the two have not been reported coexisting. A 16-month-old ex-premature boy was found unresponsive, with his tracheostomy tube dislodged. Tracheomalacia, subglottic stenosis, and a laryngeal cyst had been diagnosed shortly after birth, and the cyst was surgically treated at that time. At autopsy, a complete tracheal ring was noted in the subglottic region, above the tracheostomy site, and the tracheal diameter was markedly decreased in this area. In addition, multiloculated cysts were present at that level, arising from both anterolateral tracheal walls. These completely occluded the airway. To our knowledge, this is the first case of a combination of tracheal ring anomaly and subglottic cysts. Pediatric pathologists must be aware of iatrogenic lesions associated with care of the premature neonate, particularly as the age of viability continues to decrease.