Keywords
esophageal mucosal bridge - miniature stapler - iatrogenic tracheoesophageal fistula
Introduction
Esophageal atresia (EA) is a congenital disorder that affects approximately 1 in 2,500
neonates. In the follow-up of these patients even after successful reconstruction,
various complications sometimes occur long-term, such as dysphagia. Mucosal bridges
after EA repair have been described in the literature before, and are one of the rare
complications associated with dysphagia.[1] They should be corrected by division or resection when symptomatic. Since only a
very few reports have been published about this rare condition, there are no published
standards of treatment so far. We herein report the first use of a miniature stapler
device to divide a mucosal bridge at the cervical gastroesophageal anastomosis after
gastric interposition in the setting of a case of complex esophageal atresia.
Case Report
The patient was a 4-year-old girl born at 33 weeks of gestation with EA and a distal
tracheoesophageal fistula (TEF). She underwent surgical correction of EA on the early
day of life. After her primary repair, she developed a multitude of complications,
including anastomotic stricture and recurrent fistula. She underwent over 20 esophageal
dilatations and five esophageal stent placements, as well as a tracheostomy in an
outside hospital, before being referred to our center. We initially performed a bronchoscopic
and esophagoscopic examination under general anesthesia to investigate the condition
of esophagus. An on-table contrast esophagography showed a tracheoesophageal fistula
at the level of the upper esophagus ([Fig. 1A]). Endoscopy revealed a large tracheoesophageal fistula which was most likely iatrogenic
after stent placement with a cuffed tracheostomy tube in place, causing erosion of
the adjacent tracheoesophageal walls ([Fig. 1B]). This finding prompted us to perform a tracheoplasty and gastric interposition
using gastric pull-up procedure. After the operation, she had no symptoms and was
not taking any medications, including antacid drugs. One year after recovery, the
patient had dysphagia with solid food. Upon endoscopy, a mucosal bridge was noted
at the level of the gastroesophageal anastomosis ([Fig. 2]). This bridge was divided under endoscopic vision using a 6 mm flexible endoscope
and a 5 mm miniature stapler (JR-ST25.2.0, JustRight Surgical) at the same time. The
procedure took 25 minutes ([Video 1]). No intra- or postoperative complications were noted. Upon follow-up, she had no
more complaints with solid food.
Video 1
Division of a mucosal bridge using miniature stapler at the anastomosis after gastric
pull-up.
Fig. 1 (A) Upper and Lower esophagus was occuluded under endoscopy inserted through the gastrostomy,
and esophagography was performed. This contrast esophagography showed tracheobronchial
contrastation (arrows), raising the suspicion of an acquired tracheoesophageal fistula.
(B) The endotracheal tube was identified from esophagus (circle). Tracheoscopy and endoscopy
showed the large tracheoesophageal fistula, most likely due to compressive erosion
by pressure from the balloon of the cuffed tracheostomy tube and the esophageal stent
(circle).
Fig. 2 Endoscopic view of the mucosal bridge (white arrow) at the level of the gastroesophageal
anastomosis.
Discussion
Acquired TEF is an uncommon clinical condition, most frequently arising as sequelae
to esophageal malignancy in adult patients. In addition, iatrogenic injury to the
membraneous trachea secondary to cuffed endotracheal or tracheostomy tubes in the
presence of an in-dwelling nasogastric tube and corrosive burns have been reported
in the literature.[2] Our hypothesis for the TEF in this patient is that the combination of esophageal
stent placement and cuffed tracheostomy tube caused pressure necrosis, inflammation,
and erosion between these structures, forming a secondary large iatrogenic fistula.
Therefore, the combination of using cuffed tracheostomy tubes and stents in the upper
esophagus should be strictly avoided.
Mucosal bridge is a very rare condition and the etiology is still unclear. I can form
after an anastomosis, perhaps due to injury from indwelling catheters, contained leaks,
gastroesophageal reflux disease, inflammation, esophageal dysmotility, or a combination
of the above.[1] The mucosal bridge of this patient may have been the result of gastroesophageal
reflux from the interposed stomach into the upper pouch. Endoscopy is the method of
choice to diagnose these mucosal bridges and should be performed for patients that
exhibit continuous or progressive symptoms of dysphagia.
Some asymptomatic esophageal mucosal bridges were not treated.[3] However, in those showing symptoms, they should be divided or resected. Up to now,
various techniques and cutting devices have been employed to dissect the bridge; including
argon plasma coagulation, endoscopic knives, or endoscopic scissors.[4] Recently, treatment of an esophageal diverticulum with a 5 mm endoscopic stapler
has been reported.[5] This prompted us to use the endoscopic miniature stapler on the mucosal bridge of
this patient which was simple, quick (25 minutes operative time), and without complications.
However, the limitation of this technique is the size of the patient, because it is
necessary to insert the 5 mm stapler and the endoscope into esophagus at the same
time. In addition, an intraoperative challenge we encountered during the procedure
was to obtain a good seal for gas insufflation during flexible endoscopy with both
the endoscope and the stapler at the cervicothoracic junction. Therefore, rigid esophagoscopy
may be an alternative that does not require insufflation and therefore may be even
quicker.
Conclusion
The combination of esophageal stent placement and cuffed tracheostomy tube can cause
tracheoesophageal fistula by erosion and therefore should be avoided. Mucosal bridges
are rare but may cause dysphagia after esophageal atresia repair. They can be safely
divided endoscopically using a 5 mm miniature stapler.
