Background/Purpose: Clinical manifestations of airway obstruction in infants with repaired esophageal atresia or tracheoesophageal fistula (EA/TEF) are attributed conventionally to tracheomalacia. In the current study, the authors tested the hypothesis that a retrodisplacement of the tongue (glossoptosis), by causing a functional upper airway obstruction (obstructive apnea/hypopnea), may play a role in the pathogenesis of the respiratory problems. Methods: The records of 5 infants with repaired EA/TEF and respiratory symptoms treated by glossopexy, which serves to anchor the tongue forward, were reviewed. Prompt improvement after glossopexy in clinical manifestations, in blood gas exchange, and in weight velocity was used to establish a primary cause-effect relationship between glossoptosis and respiratory problems. Results: Before glossopexy, all infants presented with severe respiratory distress. Three infants had life-threatening events requiring recurrent or chronic airway intubation. Four infants had severe tracheomalacia, diagnosed at bronchoscopy. All infants presented obstructive apnea/hypopnea with desaturation. Body weight was below the third percentile in all infants. After glossopexy, 3 presented a marked clinical improvement associated with significant increase in mean oxygen saturation (88% ± 0.4 SE v95% ± 0.3 SE; P <.001). In another infant, glossopexy allowed permanent decannulation but did not relieve the problem during crying or excitement when obstructive hypopnea with desaturation required supplemental oxygen. After glossopexy and despite subsequent aortopexy, the last infant could not be extubated and died later of mycotic sepsis. Autopsy results showed a vascular ring. In the 4 surviving infants, glossopexy was followed promptly by significant catch-up growth and subsequent normal growth velocity (P = .03). Conclusions: Present findings support the concept that, in some infants with EA/TEF, respiratory problems are mainly caused by recurrent obstructive apnea/hypopnea, which may be treated successfully by lip-tongue adhesion procedures. Severe airway obstruction, not relieved by glossopexy, requires aortopexy to reduce the respiratory load imposed by tracheomalacia.
- Apparent life-threatening event (ALTE)
- Obstructive sleep apnea
- Pierre Robin syndrome
ASJC Scopus subject areas