Glossopexy as an alternative to aortopexy in infants with repaired esophageal atresia and upper airway obstruction

Pathogenesis of apparent life‐threatening events in infants with esophageal atresia

“…We found that the respiratory problems of the present infants, who were fed via gastrostomy, are very similar to those previously described in some infants with repaired EA during or after oral feeding. We used the term ALTE 13,14 to indicate an episode previously denominated as ''temporary cardiac and respiratory arrest,'' 1 or ''reflex apnea'' or ''reflex respiratory arrest'' or ''lifeless attacks'' or ''cyanotic periods,'' 6 or ''cyanotic attacks,'' 8 or ''life-threatening anoxic spells.'' 9 Other terms commonly used are ''death spell'' and ''blue spell.''…”

“…6,9,12 Likewise, the present association between signs of inspiratory airway obstruction with signs of expiratory airway obstruction was previously reported. 6,8,[12][13][14]17 Extrathoracic AO can be diagnosed by the presence during inspiration of retractions of neck soft parts, due to the negative pressure inside the extrathoracic airways below the obstruction. 18,19 Actually, in infants with repaired EA and AO/ALTE syndrome, the inspiratory airway obstruction was attributed to laryngeal spasm, 1,17 associated laryngomalacia, 6 respiratory infection, 8 glossoptotic pharyngeal obstruction, 12,13 or mechanical compression of a malacic intrathoracic trachea.…”

“…22 Therefore, in infants with inspiratory airway obstruction, such as the present infants with AO, grunting is a sign consistent with the diagnosis of lower airway instability, due to underventilation and/or alveolar hypoxia. [12][13][14] The presence of segmental collapse of dependent zones of the lung is not detected on conventional chest films. 20 In these areas, a subsequent episode of complete AO or an expiratory effort may result in widespread small-airway closure, sudden absorption collapse, and massive right-to-left shunting, explaining the rapid onset of ALTE in some infants with a repaired EA.…”

“…[10][11][12] Support for this concept comes from the good results obtained in a few infants with repaired EA and AO/ALTE syndrome treated by glossopexy, which stabilizes the upper airway by avoiding, to a certain extent, the falling back of the tongue. 13,14 At present, in infants with EA, there is still controversy as to whether overactive laryngeal chemoreflex, intrathoracic TC, and dysautonomic control of pharyngeal airway patency are independent or interrelated pathogenic factors. In addition, it is not well-recognized that not only oral feeding but also other factors may trigger clinical manifestations of AO/ALTE syndrome.…”

Assessment of obstructive apnea by using polysomnography and surgical treatment in patients with Beckwith-Wiedemann syndrome

“…The Japanese literature [9] described a neonate with Beckwith-Wiedemann syndrome who demonstrated temporary recovery after glossopexy. Glossopexy has been used for upper airway obstruction in Robin sequence [5] and repaired esophageal atresia [10]. The selection of surgical procedures may depend on the extent of macroglossia and associated abnormalities, especially ankylogrossia.…”