Keywords
esophageal atresia - upper pouch - perforation - iatrogenic
Introduction
Pharyngeal and esophageal perforation secondary to nasogastric (NG) or orogastric
(OG) tube placement is rare and occurs most frequently in preterm infants who have
weaker pharyngeal muscular structures and are at increased risk of iatrogenic esophageal
perforation.[1]
[2] Esophageal perforation in a full-term infant may occur with repeated attempts to
place NG, OG, or endotracheal tubes. Eklöf et al first described iatrogenic esophageal
perforation in 1968.[3] Review of the literature reveals misdiagnosed esophageal atresia (EA) in the setting
of esophageal perforation. Some infants have mistakenly undergone unnecessary thoracotomy
for the same.[1]
[4] However, esophageal perforation with EA is exceedingly rare. Perforation of the
upper pouch in tracheoesophageal fistula (TEF) with EA was reported by Wright and
Noblett, almost 35 years ago (1978).[5] Here, we report a case of pure EA that had this rare complication of perforation
of blind upper pouch of esophagus.
Case Report
A newborn baby weighing 1,400 g was transferred to our neonatal intensive care unit
on day 3 after birth with symptoms of frothing after birth, with suspected EA. The
baby was born at 32 weeks' gestation by normal vaginal delivery to a 22-year-old gravida
1, para 1 mother who went in premature labor. She had a normal prenatal ultrasound
scan. She was hemodynamically unstable and required vasopressors. Baseline hematological
investigations were normal except C reactive protein, which was 31 (N = 0–6 mg/L). To our surprise, the stiff rubber catheter (10 fr), passed to document
the atresia and for taking the X-ray, went smoothly and effortlessly for 25 cm.
But, the X-ray showed the tube ending in the right pleural cavity with right pneumothorax
and gasless abdomen suggestive of pure EA with possible iatrogenic perforation of
the upper pouch ([Fig. 1]). The catheter was removed immediately. Owing to increasing respiratory distress,
the baby was intubated and ventilator support was provided. Right-sided intercostal
drainage tube was inserted. The patient gradually improved and stabilized with ventilatory
support.
Fig. 1 X-ray showing catheter in right pleural cavity and gasless abdomen.
Cervical esophagostomy and feeding gastrostomy without thoracotomy were done. There
was 0.5 × 0.3 cm perforation at the tip of the pouch with irregular margins ([Fig. 2]). Left cervical esophagostomy was done after mobilizing upper pouch completely.
Fig. 2 Intraoperative photograph showing perforation at the tip of upper pouch.
Discussion
Pharyngeal and esophageal perforations are rare but well-known complications of NG
and OG catheter placement. Infants weighing less than 1,500 g have weaker pharyngeal
muscular structures and are at increased risk of iatrogenic esophageal perforation.[1]
[4] Neonatal esophageal perforation can present as pharyngeal or esophageal pseudodiverticulum
secondary to a contained cervical perforation (less common), pneumothorax, and perhaps
most commonly as esophageal obstruction. This results from compression of the lumen
by the mass effect created by the false passage. It is this presentation that can
be misdiagnosed as EA.[6] Unlike atresia, treatment for esophageal perforation typically is medical, with
antibiotics, parenteral or gastrostomy feedings, and tube thoracostomy for pneumothorax
or pleural effusion.[2]
[6] The differentiation of these two entities therefore is very important.
Perforation of upper pouch in EA with TEF (type C) was reported by Wright and Noblett
in 1978. They reported it as a complication of using plastic sump catheter for continuous
suction of upper pouch.[5] However, in our case, perforation may have occurred due to the use of stiff catheter
for the diagnosis of EA. The use of a stiff catheter can result in one of the following
possibilities: (i) uneventful passage into the stomach through a normal esophagus,
ruling out EA; (ii) hold up in the upper pouch confirming EA; (iii) iatrogenic perforation
of a normal esophagus producing a clinical picture that mimics EA, a false-positive
situation; and (iv) iatrogenic perforation of the upper pouch in the case of EA, with
the catheter passing into the mediastinum and pleural cavity (as in our case).[7]
Alternatives to the use of stiff catheter for diagnosis of EA are available, such
as fluoroscopic swallow study and endoscopy. The risk of aspiration pneumonitis and
adequate delineation of upper blind pouch with stiff catheter radiograph makes routine
fluoroscopic swallow study less popular.[7] Bronchoscopy may help in verification of the diagnosis, determination of fistula
level, and detection of an occult second fistula with rigid esophagoscopy, which can
further clarify the diagnosis and appears safe in experienced hands.[8] Bronchoscopy and esophagoscopy allow direct visualization of a TEF and blind-ending
esophagus.[1] However, none of these studies are without risk and possibly have greater risk than
a catheter radiograph.
Conclusion
A radiograph after placing catheter (NG) in the upper pouch is an accepted way of
diagnosing EA with TEF. The treating surgeon should be vigilant especially in a premature
infant or infant weighing less than 1,500 g, as a small force may perforate the upper
pouch due to inherent weakness of the dilated tissue and musculature.
