A bedridden 72-year-old man presented with massive hematemesis;
esophagogastroduodenoscopy (EGD) at a local clinic revealed esophageal ulcer
bleeding. After injection of diluted epinephrine, he was transferred to our
hospital and admitted to the intensive care unit for hypovolemic shock (blood
pressure, 82/47 mmHg; hemoglobin, 6.9 g/dL).
The patient’s medical history included right pneumonectomy for
pulmonary tuberculosis 8 years previously, and a tracheostomy for lung hygiene.
Chest X-ray film revealed right side pneumonectomy with tube tracheostomy. The
trachea was deviated to the right, and the tip of the tracheostomy tube
impacted the trachea at the T3 vertebral level.
Because melena and hematochezia persisted for 2 days, we repeated
EGD. One polypoid lesion with erosion and intermittent bleeding was noted in
the middle esophagus, which was obviously narrowed by external compression ([Figs. 1] and [2]).
Fig. 1 A polypoid lesion with
an erosive base was noted in the narrowed lumen of the mid esophagus.
Fig. 2 Intermittent oozing of
blood from the polypoid lesion was noted when it was touched by the
endoscope.
Esophageal erosive ulcer bleeding resulting from compression by the
tip of the tracheostomy tube was considered. A total 2.5 mL
N-butyl-2-cyanoacrylate with 2.5 mL Lipiodol was injected into the
erosion base ([Fig. 3]).
Fig. 3 N-butyl-2-cyanoacrylate
and Lipiodol was injected in the erosive base. The lesion was covered with a
crystal-like coating and hemostasis was achieved.
Subsequent chest X-ray film revealed that one vessel formed a
fistula with the middle esophagus ([Fig. 4]).
Fig. 4 One vessel (arrowheads)
formed a fistula with the mid esophagus (arrows). The tip of the tracheostomy
tube impacted the tortuous trachea, externally compressing the azygos vein and
mid esophagus.
Computed tomography (CT) showed that the fistulous vessel was the
azygos vein, terminating in the superior vena cava. After endoscopic
sclerotherapy, there was no recurrence of bleeding. The patient was discharged
1 month later and has had an uneventful course for the past 2 years.
Both tracheoesophageal fistula and tracheoarterial fistula are not
uncommon but fatal complications after tracheostomy [1]
[2]. To the best of our knowledge,
there are no reports of azygoesophageal fistula caused by compression of the
tracheostomy tube. In our case, the possible pathogenesis of the
azygoesophageal fistula was that the trachea was deviated due to the previous
pneumonectomy, and the tip of the tracheostomy tube impacted the wall of the
tortuous trachea and pushed the azygos vein towards the mid esophagus.
N-butyl-2-cyanoacrylate is widely used not only in bleeding gastric
varices but also in peptic ulcer hemorrhage or Dieulafoy lesions
[3]
[4]. N-butyl-2-cyanoacrylate
injection has been recommended as a last resort before surgery
[5]. This case suggests that clinicians should pay
attention to the late complications of tracheostomy, such as fistula between
the trachea, esophagus, and major vessels. Furthermore, the bleeding from an
azygoesophageal fistula can be defined and safely occluded by sclerotherapy
with Lipiodol.
Endoscopy_UCTN_Code_TTT_1AO_2AD
