Paediatric Respiratory Reviews 19 (2016) 10–15

Contents lists available at ScienceDirect

Paediatric Respiratory Reviews

Mini-symposium: Oesophageal Atresia and Tracheo-oesophageal Fistula

Surgical management of oesophageal atresia

Warwick J. Teague 1,2,3, Jonathan Karpelowsky 4,5,*
1
Academic Paediatric Surgeon, Department of Paediatric Surgery, The Royal Children’s Hospital, Melbourne, VIC, Australia
2
Clinical Associate Professor, Department of Paediatrics, The Royal Children’s Hospital, Melbourne, VIC, Australia
3
Honorary Fellow, Surgical Research Group, Murdoch Children’s, Melbourne, VIC, Australia
4
Paediatric Surgeon, Department of Paediatric Surgery, The Children’s Hospital at Westmead, Locked Bag 4001, Westmead 2145, Sydney, NSW, Australia
5
Senior Lecturer, Discipline of Paediatrics & Child Health, Sydney Medical School, University of Sydney, Sydney, NSW, Australia

EDUCATIONAL AIMS

The reader will come to appreciate how:

1. To understand the rationale for preoperative work up in oesophageal atresia

2. To describe the surgical correction of oesophageal atresia
3. To review the role of thoracoscopic surgery in oesophageal atresia
4. To describe surgical options to correct long-gap oesophageal atresia
5. To review early post-operative complications.

A R T I C L E I N F O S U M M A R Y

Keywords: There have been major advances in the surgery for oesophageal atresia (OA) and tracheo-oesophageal
Oesophageal atresia
fistula(TOF) with survival now exceeding 90%. The standard open approach to OA and distal TOF has been
Long Gap oesophageal atresia
well described and essentially unchanged for the last 60 years. Improved survival in recent decades is
Tracheo-oesophageal fistula
most attributable to advances in neonatal anaesthesia and perioperative care. Recent surgical advances
include the use of thoracoscopic surgery for the repair of OA/TOF and in some centres isolated OA,
thereby minimising the long term musculo-skeletal morbidity associated with open surgery. The
introduction of growth induction by external traction (Foker procedure) for the treatment of long-gap OA
has provided an important tool enabling increased preservation of the native oesophagus. Despite this,
long-gap OA still poses a number of challenges, and oesophageal replacement still may be required in
some cases.
ß 2016 Elsevier Ltd. All rights reserved.

INTRODUCTION presents unique surgical challenges, many of which are discussed

The first successful repair of oesophageal atresia (OA) and
tracheo-oesophageal fistula (TOF) was performed in 1941:
Cameron Haight ligated the TOF prior to an end-to-end oesopha-
geal anastomosis through a left extrapleural approach [1]. In the
subsequent 75 years the overall survival has improved to exceed
90%, with mortality now usually associated with prematurity and/
or cardiac comorbidity [2,3]. Despite improved survival, OA still
* Corresponding author. Department of Paediatric Surgery, The Children’s
Hospital at Westmead, University of Sydney, Locked Bag 4001, Westmead 2145,
Sydney, NSW, Australia. Tel.: +61 2 9845 3341; fax: +61 2 9845 3180.
E-mail address: jonathan.karpelowsky@health.nsw.gov.au (J. Karpelowsky).
here. This review focuses on the surgical management of OA and
any associated TOF, highlighting the preoperative investigations,
timing of surgery, surgical approaches, surgical complications and
challenges of ‘long-gap’ OA.
PREOPERATIVE ASSESSMENT
The two primary goals of preoperative assessment in the
patient with a clinical diagnosis of OA are:
1. Confirmation of the diagnosis;
2. Identification of associated anomalies with immediate manage-
ment implications for the planned oesophageal atresia surgery.

http://dx.doi.org/10.1016/j.prrv.2016.04.003
1526-0542/ß 2016 Elsevier Ltd. All rights reserved.

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 W.J. Teague, J. Karpelowsky / Paediatric Respiratory Reviews 19 (2016) 10–15 11

Table 1
Frequency of associated anomalies in OA (Data extracted from Burge et al. 2013 [4])

Associated Anomaly All OA OA with distal TOF Other OA subtypes

Any 52% 54% 44%

Vertebral 7% 6% 16%
Anorectal 11% 6% 40%
Cardiac 34% 36% 28%
Renal 9% 8% 16%
Limb 11% 14% 3%
VACTERL 13% 12% 24%
Chromosomal 3% - -

As more than half of OA patients have an associated anomaly

(Table 1), [4] additional systems screening for anomalies is
ordinarily deferred until after primary OA surgery. Exceptions to
this are outlined briefly below.
The clinical diagnosis of OA is made when a 10 French (Fr)
tube arrests in the upper oesophagus approximately 10-12 cm
from the lips. Tubes of narrower calibre may give the false
impression of tube passage by coiling in the dilated upper
oesophageal pouch, or rarely traversing the trachea and TOF to
enter the stomach [5,6]. A diagnosis of OA is then confirmed with
a plain chest x-ray to show the arrested 10 Fr tube within the
relative lucency of the dilated upper oesophageal pouch. An
accompanying distal TOF is indicated by the presence of
gastrointestinal gas.
Particular attention is paid to marked stomach dilatation,
which may reflect preferential ventilation of the stomach via the
TOF, or concomitant duodenal atresia with a ‘double bubble’. In
either setting, emergency distal TOF ligation is indicated to
prevent the rare but morbid complication of gastric perforation
[7]. Conversely, a ‘gasless abdomen’ on preoperative x-ray raises
the possibility of either pure OA or OA with a proximal TOF,
and should prompt further investigation as detailed below. The
chest x-ray should also be assessed for evidence of early
complications (e.g. pulmonary aspiration, intubation) or associ-
ated anomalies (e.g. abnormalities of the cardiac silhouette,
vertebrae and ribs).
A preoperative echocardiogram is undertaken to define any
associated major congenital heart disease (CHD), particularly duct-
dependent lesions which may necessitate particular anaesthetic
management or prior cardiac surgery. In addition, an echocardio-
gram may demonstrate vascular anomalies relevant to operative
decision-making, most notably a right-sided aortic arch (RAA)
which is present in approximately 4% of OA cases [8]. The
importance of routine preoperative echocardiography is highlight-
ed by expert commentators, [5,6] whilst others present data to
moderate this stance, [9,10] showing preoperative echocardiogra-
phy findings of CHD and/or RAA seldom alter the operative plan,
including the choice of right vs left thoracotomy in cases with a
known RAA [8,10].
The role of routine preoperative laryngotracheobronchoscopy
(LTB) in OA patients remains a matter of debate, [11] with only
43-60% of contemporary paediatric surgeons routinely using
preoperative LTB in this setting [12,13]. Advocates cite preopera-
tive LTB findings which may impact management in 21-45% of OA
patients, most notably unusual fistula position (Figure 1) and
tracheobronchial tree anomalies, and less commonly laryngeal
clefts or subglottic stenosis [14–16]. The presence of significant
tracheomalacia may alert clinicians to the need for non-invasive
support following extubation. The benefit of preoperative LTB is
maximal in newborns with suspected pure OA due to a ‘gasless
abdomen’ on x-ray. In this OA subgroup, LTB reveals a proximal
TOF in 20-33% with resultant change in management [14–17].
An upper pouch oesophagogram may augment LTB to identify a
Figure 1. Bronchoscopy of a distal tracheo-oesophageal fistula.
‘near-missed’ proximal TOF, [18] but is highly dependent on local
radiology expertise. Preoperative endoscopic intubation of the TOF
with the aim facilitating surgical repair, Fogarty balloon TOF
occlusion to aid ventilation and selective trans-tracheal gastric
drainage have been described [11,15,16,19].
Routine screening investigations for other associated anoma-
lies would include renal ultrasound, spinal ultrasound and sacral
x-ray. These are ordinarily not performed preoperatively, except a
renal ultrasound in the newborn who has not voided (OA is
uncommonly associated with renal agenesis), [6] or urgent
genetic testing where an undiagnosed lethal syndrome is
suspected, e.g. Trisomy 13 (Patau) or 18 (Edwards).
OPEN SURGERY
With rare exceptions, OA surgery is not an emergency and can
be deferred whilst preoperative investigations are obtained and an
appropriately skilled anaesthetic and surgical team assembled.
Indications for emergency OA surgery are limited to a markedly
dilated stomach at risk of perforation, or the premature infant with
evolving Respiratory Distress Syndrome (RDS) in whom preferen-
tial TOF ventilation is a significant contributor to a deteriorating
ventilatory status. Two recent studies report increased complica-
tions in OA patients undergoing surgery within 24 hours of birth or
after-hours [20,21]. Selection biases aside, these studies highlight
the benefits of in-hours OA surgery, which may be safely deferred
beyond 24 hours of life.
The goals and key steps of the open approach have remained
largely unchanged over decades, and are summarised below.
Specific goals and operative strategies for other OA variants, e.g.
long-gap OA, are described elsewhere in this review.
Key operative goals
1. TOF ligation to prevent further soiling of the tracheobronchial
tree with stomach contents and restore the ventilation
dynamics of the intact trachea.
2. Restoration of oesophageal continuity, which can be deferred
if the child’s status is poor at completion of TOF ligation.

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12 W.J. Teague, J. Karpelowsky / Paediatric Respiratory Reviews 19 (2016) 10–15
Key operative steps
1. Right posterolateral thoracotomy, with attention to a muscle-
sparing technique to reduce the musculoskeletal sequelae of
thoracotomy in infancy [22].
2. Extrapleural approach to the posteromedial mediastinum, to
expose the azygos vein (typically, but not universally, ligated
and divided in continuity) and the TOF. TOF dissection (Figure 2)
is minimised with an aim of preserving its neurovascular supply,
and proceeds to expose the junction with the trachea in
readiness for TOF ligation.
3. TOF ligation is undertaken, with either an absorbable or non-
absorbable suture. Prior to division of the ‘fistula’, it is important
ensure neither a major bronchus nor aorta have been mistakenly
dissected as iatrogenic injuries to each have occurred. Some
advocate peroperative tracheoscopy to optimise TOF dissection and
ligation, [19] but this technique is not yet been widely adopted.
4. Upper oesophageal pouch dissection (Figure 3) optimally
commences prior to TOF ligation. Downward pressure by the
Figure 2. Dissection of trachea-oesophageal fistula.
Figure 3. Dissection of Upper blind ending pouch in oesophageal atresia.
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Figure 4. Completed thoracoscopic anastomosis.
anaesthetist on the 10Fr oesophageal tube indicates the
approximate position of the upper pouch, allowing unexpected
positions to be taken into account at TOF ligation. Following TOF
ligation, upper pouch dissection proceeds until adequate length
is obtained or the pouch is maximally dissected. Sharp and
careful dissection of the plane between the trachea and pouch is
the key to avoiding tracheal injury during this step. Finally, the
upper pouch is incised in readiness for anastomosis.
5. Oesophageal anastomosis (Figure 4) with fine interrupted
sutures begins with the back wall, following which a trans-
anastomotic nasogastric tube is passed prior to completion of
the anastomosis of the front wall.
THORACOSCOPIC SURGERY
The benefits of minimally invasive surgery over thoracotomy
for reducing pain, scars and long term musculoskeletal deformities
including scoliosis have been well documented [23–25]. The first
thoracoscopic repair of an isolated OA was undertaken in 1999,
[26] and OA with distal TOF in 2000 [27]. Since then, a number of
larger multi-institutional series have validated the thoracoscopic
technique to be equivalent to open surgery [28–30].
The patient is positioned semi prone with the right side
elevated by 30 degrees. Standard endotracheal intubation is used
with no need for endobronchial isolation. Three 3 mm ports are
used, although some surgeons use a 5 mm port to facilitate use
of an endoclip. A pneumothorax of 3-5 mmHg facilitates lung
collapse and visualisation, and a fourth port may be used should
lung retraction become necessary. Port placement is crucial due to
the limited working space afforded by the neonatal thorax.
As in open surgery, the first step is to use the azygos vein and
vagus nerve to identify the TOF. The azygos vein is often divided
(energy device, clips or ties), but may be preserved. The TOF is
dissected cranially to its junction with the membranous trachea,
prior to being ligated flush with the trachea using either a suture or
endoclip. Attention is then turned to the upper pouch, which is
identified and dissected with the aid of gentle pressure on the 10 Fr
tube in the upper pouch. The anastomosis is technically challeng-
ing, and performed using interrupted sutures secured by
intracorporeal ties. Once complete, a chest tube may be left
through one of the port site incisions.
Technical benefits of the thoracoscopic approach include
improved visualisation of the entire hemithorax and excellent
magnification [31]. Approaching the fistula perpendicularly allows
 W.J. Teague, J. Karpelowsky / Paediatric Respiratory Reviews 19 (2016) 10–15
dissection in situ with localisation of the point of entry into the
membranous part of the trachea. Dissection of the upper pouch is
also improved, with straightforward ability to extend high up into
the neck if required.
Despite the stated benefits and comparability to the open
approach, the highly skilled nature of thoracoscopic OA surgery has
limited its wider adoption [24]. A recent survey of paediatric
surgeons noted open thoracotomy was the preferred approach by
94% of surgeons, [12] albeit this number is reduced to only 50%
when surveying a cohort of surgeons with an interest in minimally
invasive surgery [13].
Concerns have been raised regarding the physiological
stresses experienced by the neonate during thoracoscopic repair,
especially hypercapnoea resulting in acidosis and cerebral
hypoperfusion [32]. Recent infrared spectroscopy data however,
goes some way to addressing these concerns by showing
hypercapnoea during thoracoscopic OA surgery is not associated
with cerebral hypoxia [33].
LONG-GAP OESOPHAGEAL ATRESIA
The surgical management of long-gap OA still remains a
significant challenge [34–36]. Currently, there is no consensus on
the definition of long-gap OA. Many discussions have focussed only
on pure OA, [37] although a recent meta-analysis confirms many
long-gap OA occur together with a distal TOF [38]. Greater
consensus exists to support prioritising the retention of the native
oesophagus, rather than proceeding primarily with oesophageal
replacement. Replacement is however, an important option for a
select cohort of OA patients, in whom ongoing attempts to retain
the native oesophagus are deemed futile and likely detrimental to
the child’s wellbeing [39,40].
Various methods have been developed to overcome the
technical difficulties of long-gap OA. Methods to preserve the
oesophagus include delayed primary anastomosis and lengthening
procedures such as circular myotomy, oesophageal flap, Foker
procedure (traction suture oesophageal lengthening) and Kimura
technique (multistage extra-thoracic oesophageal elongation).
Alternatively, oesophageal replacement may be performed with
conduits, including gastric tube, gastric transposition, small bowel
or colonic interposition [41].
Delayed primary anastomosis involves creating a gastrostomy
to facilitate feeding at or soon after birth. At this time, a ‘gap study’
may be performed to assess the length of the gap, e.g. dilator is
passed via the stomach into the distal oesophageal pouch and with
a large firm tube in the proximal oesophagus a gap is measured
using fluoroscopy. The gap length is typically expressed as ‘number
of vertebral bodies’. Many surgeons consider a gap exceeding two
vertebral bodies to be difficult to anastomose primarily, i.e. ‘long-
gap OA’. In this instance, the child is fed via gastrostomy and the
upper pouch managed by continuous aspiration of saliva. When
performed, the anastomosis can be achieved by open or thoraco-
scopic approach, with or without adjuncts to maximise length, e.g.
upper pouch flaps or myotomies [31,42].
Since first described in in 1997, [43] the Foker and other
traction-based procedures have gained popularity [44–48]. This
staged procedure involves primary mobilization of the oesopha-
geal ends prior to placement of and externalisation of traction
sutures. Tension on the externalised sutures is then gradually
increased over days to encourage elongation of the upper and
lower pouches to narrow the ‘gap’. Whether traction induces true
growth in the oesophageal ends, or only stretch, remains
controversial [23]. Once sufficient length has been achieved a
primary oesophageal anastomosis is undertaken. The placement of
traction sutures and subsequent anastomosis can be undertaken
by an open or thoracoscopic approach [46].
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13
When oesophageal preservation is not possible, several
replacement methods are available [36]. Gastric ‘pull up’ involves
mobilising the entire stomach on the right gastric and right gastro-
epiploic arteries into the posterior mediastinum and suturing the
fundus to the proximal oesophagus [49]. Conversely, a gastric
‘tube’ involves tubularisation of the greater curve of the stomach
leaving a smaller but functional stomach. The gastric tube is then
translocated into the thorax on the right gastro-epiploic vessels
[41]. Finally, colonic [50] and jejunal [51] interposition involve
isolating a segment of intestine with its mesenteric pedicle, and
‘interposing’ this conduit between the proximal oesophagus and
stomach. Despite each of these replacement options providing a
reliable conduit, the surgery is associated with a high incidence of
complications (10-45%) including mortality in 4-5% [41,52]. No one
conduit has proven to be superior, rather familiarity with the
techniques and local expertise determine success.
EARLY POSTOPERATIVE COMPLICATIONS
The three early postoperative complications of OA surgery with
greatest relevance to the patient’s medium and long-term outcome
are: anastomotic leak (3-20%), anastomotic stricture (39-57%) and
recurrent TOF (3-7%); cited incidences coming from recent British
and German multicentre prospective cohort studies [53,54].
Anastomotic Leak
Anastomotic leaks occur in 3–20% of OA patients [53–56].
Reported incidences may reflect local utilisation of ‘routine’
contrast studies to scrutinise the anastomosis, e.g. on the 5th
postoperative day, rather than technical skill. The key risk factor for
leakage is anastomotic tension [57]. Major leaks occur in 3-5% and
manifest early (<48 hours) with acute deterioration, including
tension pneumothorax and sepsis. Emergency chest tube decom-
pression is effective to control the associated pleural soiling, and in
many cases no other operative intervention is required. Re-do
thoracotomy, with or without oesophagostomy, is an uncommon
event, reserved for complete anastomotic disruption, persistent
soiling or inability to control pneumothoraces [5,55,56].
Minor leaks are more common, contained and typically noted
on ‘routine’ contrast studies in otherwise asymptomatic children.
Minor leaks seldom require specific management, and can be
expected to close spontaneously following a brief deferral of oral
feeds [55]. The use of glycopyrolate, which reduces saliva
production, has been suggested as an adjunct to encourage closure
[58]. The key long-term sequelae of anastomotic leakage include
an increased risk of troublesome stricture formation or recurrent
TOF [55,56].
Anastomotic Stricture
Anastomotic stricture is common, occurring in approximately
one third of OA patients. The key risk factors for stricture formation
are anastomotic tension, gastro-oesophageal reflux and previous
anastomotic leak [55,57,59–61]. Serial dilatation is a safe and
effective treatment, be that using balloon or bougie techniques
[57,60–62]. Both approaches are associated with a low risk of
perforation (<2% per dilatation episode), albeit that the vast
majority of perforations are managed non-operatively [59,60,62].
Recurrent TOF
Recurrent fistula formation is an uncommon but morbid
complication, which may present with recurrent chest infections
and acute life-threatening events and prove difficult to diagnose
[63]. Treatment may be by an open or endoscopic approach, the
14 W.J. Teague, J. Karpelowsky / Paediatric Respiratory Reviews 19 (2016) 10–15
latter including thermal ablation using laser or diathermy followed
by occlusion with fibrin or Cyanoacrylate glue [64,65]. The role of
endoscopic management remains a matter of controversy. A recent
systematic review found open surgery to be superior to endoscopy
with lower morbidity, fewer treatment episodes and greater
success [66]. However, the less invasive nature of endoscopic
management makes this a suitable option for a well-selected
patient.
Vocal cord paresis
Vocal cord paresis (VCP) is reported in approximately 3-4% of
patients post OA surgery [67,68]. The presumed aetiology is
intraoperative injury to the recurrent laryngeal nerve. This
notwithstanding, congenital VCP has also been reported in OA
patients, highlighting a benefit of preoperative LTB. Whilst VCP
cases manifest overtly with early stridor or failed extubation,
others present diagnostic difficulties [68,69]. Unilateral VCP
is managed expectantly and may resolve spontaneously,
whereas bilateral VCP requires operative management with
tracheostomy [68].
LATE POSTOPERATIVE COMPLICATIONS
The interplay of gastro-oesophageal reflux and/or tracheoma-
lacia with OA is a cause of significant morbidity, including acute
life-threatening events. Medical and surgical management each
have a role to mitigate such complications [70,71]. These and other
long-term aerodigestive complications and their management are
discussed elsewhere in this mini-symposium, and are the subject
of a recent meta-analysis [72].
FUTURE RESEARCH DIRECTIONS
Future research in surgical therapy for oesophageal atresia
needs to focus on several key areas:
1. There is a need for standardisation of the definition of ‘long-gap
OA’ and taxonomy of the procedures used to address this
particular OA subtype. This advance would facilitate compara-
tive and collaborative research investigating surgical manage-
ment of this challenging OA subtype.
2. There should be evaluation of the long-term complications of
open compared with thoracoscopic surgery, to evaluate whether
the stated benefits of a technically more difficult but less
invasive technique are warranted.
3. There is a need for investment in basic research to overcome
barriers currently preventing creation of a biological conduit
suitable for use in oesophageal replacement.
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