The surgical management of non-malignant aerodigestive fistula

Abstract

Background
Acquired aerodigestive fistula (ADF) are rare, but associated with significant morbidity. Surgery affords the best prospect of cure. We present our experience of the surgical management of ADFs at a specialist unit, highlighting operative techniques, challenges and assess clinical outcomes following intervention. We also illustrate findings of a Hospital Episodes Statistics search for ADFs.

Methods
A prospectively-maintained database was searched to identify all patients diagnosed with an ADF who were managed at our institution. Of 48 patients with an ADF, eight underwent surgical intervention.

Results
Four patients underwent an exploration of the ADF with primary repair of the defect. Two of these patients had proximal ADFs, amenable to repair through a neck incision, and two required a thoracotomy. Two patients suffered fistulae secondary to endoscopic therapy and underwent oesophageal exclusion surgery, with subsequent staged reconstruction. Two patients with previous Tuberculosis had a lung segmentectomy and lobectomy respectively, and a further patient in remission after treatment for lymphoma underwent oesophageal resection with synchronous reconstruction. Three patients suffered a complication, with one post-operative mortality. The remaining seven patients all achieved normal oral alimentation, with no evidence of ADF recurrence at a median follow-up of 32 months.

Conclusions
Surgery to manage ADFs is effective in restoring normal alimentation and alleviates soiling of the airway, with a very low risk of recurrence. Several operative techniques can be utilised dependent on the features of the ADF. Early referral to specialist units is advocated, where the expertise to facilitate the complete management of patients is present, within a multi-disciplinary setting.

Full text

R E S E A R C H A R T I C L E Open Access
The surgical management of non-
malignant aerodigestive fistula
Yassar A. Qureshi
1*
, M. Muntzer Mughal
1
, Sheraz R. Markar
2
, Borzoueh Mohammadi
1
, Jeremy George
3
,
Martin Hayward
4
and David Lawrence
4
Abstract
Background: Acquired aerodigestive fistula (ADF) are rare, but associated with significant morbidity. Surgery affords
the best prospect of cure. We present our experience of the surgical management of ADFs at a specialist unit, highlighting
operative techniques, challenges and assess clinical outcomes following intervention. We also illustrate findings of a Hospital
Episodes Statistics search for ADFs.
Methods: A prospectively-maintained database was searched to identify all patients diagnosed with an ADF who were
managed at our institution. Of 48 patients with an ADF, eight underwent surgical intervention.
Results: Four patients underwent an exploration of the ADF with primary repair of the defect. Two of these patients had
proximal ADFs, amenable to repair through a neck incision, and two required a thoracotomy. Two patients suffered fistulae
secondary to endoscopic therapy and underwent oesophageal exclusion surgery, with subsequent staged reconstruction.
Two patients with previous Tuberculosis had a lung segmentectomy and lobectomy respectively, and a further patient in
remission after treatment for lymphoma underwent oesophageal resection with synchronous reconstruction. Three patients
suffered a complication, with one post-operative mortality. The remaining seven patients all achieved normal oral
alimentation, with no evidence of ADF recurrence at a median follow-up of 32 months.
Conclusions: Surgery to manage ADFs is effective in restoring normal alimentation and alleviates soiling of the airway, with
a very low risk of recurrence. Several operative techniques can be utilised dependent on the features of the ADF. Early
referral to specialist units is advocated, where the expertise to facilitate the complete management of patients is
present, within a multi-disciplinary setting.
Keywords: Aerodigestive fistula, Tracheo-oesophageal fistula, Oesophageal cancer, Oesophageal surgery
Background
Surgical intervention affords the best prospect of
long-term cure of aerodigestive fistulae (ADF). Although
several operative techniques can be used to treat this de-
bilitating condition, they can only be utilised in selected
patients owing to both the underlying diagnosis and the
risks associated with such surgery [1–4]. However, with
ADFs becoming an increasing health problem, with
improving diagnosis and evolving peri-operative care, it
is likely that surgery will play a more important role in
the management of ADFs.
The choice of operative technique to treat ADFs is
dependent on several factors. However, the most import-
ant facet relates to the underlying oesophageal or airway
disease, which determines the state of tissue and its
amenability to repair and future surveillance, if required
[1,2,5,6]. Patients often present in a physiologically
challenged state owing to the nature of the disease, and
many will not be candidates for surgery. However, fo-
cused pre-operative intervention and nutritional support
may enable some patients to proceed to surgery. For
these reasons, a multi-disciplinary (MDT) approach is
necessary, and underscores why these patients should be
managed in dedicated centralised units. The range of op-
erations include resection and reconstruction, exclusion
and bypass of the affected segment of oesophagus, and
exploration and repair of the ADF. The expertise of head
* Correspondence: yassar.qureshi.17@ucl.ac.uk
1
Department of Oesophago-Gastric Surgery, University College London
Hospital, 250 Euston Road, London NW1 2BU, UK
Full list of author information is available at the end of the article
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International License (http://creativecommons.org/licenses/by/4.0/), which permits unrestricted use, distribution, and
reproduction in any medium, provided you give appropriate credit to the original author(s) and the source, provide a link to
the Creative Commons license, and indicate if changes were made. The Creative Commons Public Domain Dedication waiver
(http://creativecommons.org/publicdomain/zero/1.0/) applies to the data made available in this article, unless otherwise stated.
Qureshi et al. Journal of Cardiothoracic Surgery (2018) 13:113
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and neck, thoracic and oesophago-gastric surgeons is re-
quired to manage these patients.
In this study, we present our experience of surgical
intervention in patients diagnosed with an ADF. We
explore the background leading to the development of
an ADF, and relate how this can impact on the nature of
surgery performed. Furthermore, we describe the opera-
tive technique, challenges and outcomes following inter-
vention. We review the pertinent literature to enable an
evidence-based approach to the surgical management of
ADFs. We also illustrate findings of a Hospital Episodes
Statistics (HES) search for ADFs, highlighting the
challenges of diagnosis, management and reporting in
contemporary practise.
Methods
We interrogated a prospectively-maintained database to
identify patients diagnosed with an ADF and managed at
our institution between January 2005 and January 2017.
A total of 48 patients with an ADF were identified, of
whom eight patients have undergone surgery to treat
their fistula. All patients were discussed at a specialist
MDT where a consensus on optimal management was
reached. Of the 40 patients managed non-surgically, 31
were treated with endoscopic intervention (oesophageal
or tracheal stent), mostly owing to the presence of ad-
vanced malignancy not amenable to curative treatment.
Endoscopic treatment facilitated an alleviation of re-
spiratory soiling, whilst allowing oncological treatment
to be commenced. A further seven patients were man-
aged in palliative setting after presenting in e xtremis,
and two patients with very small asymptomatic ADFs
were managed conservatively with regular surveillance.
Follow-up refers to time from diagnosis of ADF (or
underlying disease where specified) to last clinical en-
gagement or death. Median follow-up was 32 months.
Local ethical approval for retrieval and use of clinical
data for this study was granted.
Operative technique
When considering surgery as treatment for ADF, several
factors should be specifically assessed for. It is impera-
tive that a careful search for malignancy is performed
prior to surgery, particularly as many patients will have a
preceding history of proximal oesophageal squamous
cell carcinoma (SCC) treated with chemo-radiotherapy.
If active malignancy is present in the context of an ADF,
this represents locally advanced disease with poor out-
come, rarely amenable to curative surgical intervention.
In these patients, endocopic treatment should be considered
to alleviate symptoms, coupled with chemo-radiotherapy if
appropriate. The physiological state of the patient must also
be thoroughly assessed, to ensure that the risks of major
morbidity and mortality after surgery are minimised, and
thatthepatientwouldbeabletorecoverfromsuchinter-
vention. Patients should be carefully optimised, and where
indicated, the pre-operative placement of a feeding jejunost-
omy and a venting gastrostomy to improve the nutritional
and metabolic state, and to minimise continued soiling of
the airway, should be performed.
Once a patient is deemed to have an ADF curable by
surgery, secondary factors relating to the ADF and
surrounding tissue become important considerations. A
larger defect, a history of previous local radiotherapy and
endoscopic intervention are all factors which make sur-
gery more challenging. Also, the location of the ADF is
important, as more proximally sited fistulae are amenable
to repair through a neck incision, yet for distal ADF a
thoracotomy is mandated, carrying a greater risk of major
morbidity and mortality. If there has been significant local
contamination, then it may be prudent not perform a
synchronous reconstruction, as the likelihood of an anas-
tomotic dehiscence increases. In these patients, a delayed
reconstruction confers improved chances of better reco-
very. However, given the heterogenous aetiology of ADF,
each case should be considered with a view to an indivi-
dualised treatment plan.
At induction, for tracheo-oesophageal fistulae (TOF),
it is important that the endotracheal tube balloon is sited
distal to the fistula. This will avoid inadvertent damage
to the cuff whilst dissecting and exposing the fistula, and
negate the possibility of ventilatory embarrassment
intra-operatively. Furthermore, this manoeuvre mini-
mises further contamination of the respiratory tract by
manipulation of the affected structures during surgery.
ADF exploration and repair
This may involve either an incision in the neck for proximal
fistulae, or a thoracotomy for more distal ADFs. In the neck,
dissection must proceed to mobilise the thyroid with careful
identification and preservation of the recurrent laryngeal
nerves and parathyroid glands. The oesophagus should be
circumferentially mobilised, as this manoeuvre will allow the
pharyngo-laryngeal complex to be gently pulled superiorly
and away from the thoracic inlet, to provide good access to
the fistula. Once the fistula has been identified, it can be dis-
sected free and a primary repair of the oesophagus and tra-
chea with absorbable sutures can be performed. It is critical
that the fistula is accessible from both sides of the neck to
ensure complete control of the airway during the repair,
whilst also facilitating a pedicled strap muscle interposition
flap. This reinforces the repair by providing a physical bar-
rier between the two suture lines.
In the thorax, a similar approach is used with an inter-
costal flap which is carefully prepared at the time of
thoracotomy. Once the fistula has been identified, again,
it is dissected free and a primary repair performed, with
the intercostal flap placed between the suture lines.
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Exclusion
Exclusion surgery involves isolating the oesophagus from
alimentary tract continuity, both proximal and distal to
the fistula. This involves an incision in the neck to ac-
cess the proximal oesophagus, where, once circumferen-
tially mobilised, it is transected above the fistula and
brought to the skin as an oesophagostomy. If the fistula
is very proximal, then the superior oesophagus may be
left in situ, and a large T-tube placed in the lumen with
the distal limb of the tube brought to the skin.
Next, a laparotomy is performed where the oesohago-
gastric junction (OGJ) is mobilised and the stomach
transected below this, from the lesser curve through to
thefundus.Thismanoeuvreexcludestheoesophagusfrom
the GI tract entirely, whilst preserving the majority of the
stomach for future reconstruction. The small stomach
remnant attached to the OGJ is brought to the abdominal
wall, where a generous gastrostomy is fashioned. This al-
lows retrograde access to the excluded oesophagus, for both
endoscopic surveillance and therapy, and facilitates venting
of oesophageal mucous.
Our unit policy is to defer reconstruction as a second,
staged procedure. This allows the patient a period of re-
covery, whilst respiratory and nutritional optimisation
continues. Furthermore, by fashioning an anastomosis at
the index operation in a potentially contaminated surgi-
cal field, there is a higher chance of a leak. If this were
to occur, there is substantial risk of fistula recurrence.
Where possible, the stomach is used a conduit, and is
brought to the neck through the retrosternal space, thus
avoiding the need for a repeat thoracotomy. If there is
insufficient proximal oesophagus, the stomach may be
anastomosed directly to the inferior pharyngeal
constrictors.
Resection
This is normally reserved for large or recurrent fistulae.
For proximally sited ADF - those affecting the trachea,
this will involve resection of the oesophagus, via a trans-
thoracic approach. The fistula is identified, and the
oesophagus dissected away around it. However, the
oesophageal tissue intimately involved with the fistula is
left in situ, thus avoiding direct dissection of the trachea
and minimising the risk of an air leak. The tracheal defect
with the overlying oesophageal tissue is then primarily
closed, with the latter acting as a buttress reinforcing the
tracheal repair. Typically, a gastric conduit is utilised for
reconstruction, necessitating a laparotomy.
For more distal ADF, those affecting the bronchus inter-
medius and more distal, a thoracotomy is performed to
identify the fistula. A segmentectomy or lobectomy of the
lung can be performed, dependent on the size of the de-
fect and the quality of the surrounding parenchyma. Thus,
the affected distal airway and the fistula are excised en
bloc. The oesophageal defect can be repaired primarily,
utilising an intercostal flap to reinforce the repair, or an
oesophageal resection is performed if the defect is very
large and unlikely to heal. In these instances, given the
anastomosis will be at a distinct site from the ADF, a syn-
chronous reconstruction can be performed safely.
In our experience, tracheal resection is a very challen-
ging operation, with the risk of significant short and
long-term complications [2,3]. Owing to the limited
vascularity of the trachea, healing, particularly in this co-
hort of patients, may be protracted, necessitating pro-
longed mechanical ventilation. Thus, we have preferred
to avoid such an operative intervention. However, for
very large TOFs, or those where a circumferential injury
to the trachea is present (such as cuff related fistulae), or
where other intervention has failed, tracheal resection
and reconstruction may be indicated. Mathisen et al pro-
vide an operative description and experience of this
technique [3].
Results
Preceding history and previous intervention
The median age at diagnosis of ADF was 56 years (range
29–73 years). Three patients had a previous diagnosis of
oesophageal malignancy; all were treated with chemo-
and/or radiotherapy, and one with surgical resection in
addition. Two patients had a prior diagnosis of Tubercu-
losis (TB) and had received anti-microbial therapy in the
past, and one patient previously had surgical interven-
tion following a post-emetic oesophageal leak. In two pa-
tients, no obvious cause of ADF was identified, likely
representing congenital fistulae that had persisted into
adulthood. Of these cases, two patients (1 and 6) devel-
oped oesophageal strictures after their initial treatment.
Patient 1 had undergone several balloon dilations and
stent placements, with the stent subsequently eroding
into the airway (Fig. 1). Similarly, patient 6 also received
a stent which directly caused the fistula. Table 1summa-
rises key patient factors.
ADF characteristics
The two patients with an unknown cause of fistula had a
very long history of symptoms, and had been managed
in the community with a diagnosis of asthma (Table 2).
The median time to ADF development for the three pa-
tients with a malignancy was 15 months (range 3–21),
with the shortest time affecting a patient who had an
oesophageal lymphoma (Patient 7). She had a complete
response to chemotherapy, with a residual fistula persist-
ing (Fig. 2). Both patients with TB had a long interval
after curative medical therapy, although they had sug-
gestive symptoms for some time prior to referral. Most
patients presented with recurrent chest infections and
symptoms suggestive of aspiration. Of these, one patient
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(4) presented with acute respiratory failure owing to
overwhelming infection caused by aspiration. Of interest,
he had a fistula affecting the very proximal trachea
(Fig. 3).
The size of the fistula ranged from 3 to 16 mm, with
the larger defects affecting those who had a prior diag-
nosis of carcinoma or who underwent surgical treat-
ment. The location of the fistula in relation to the airway
too was variable, reflecting the site of underlying disease.
Those thought to be congenital were very proximal.
Those secondary to TB were both distal, involving the
smaller bronchi and lung parenchyma at the original
Ghon focus. Patient 6 presented with Boerhaave’s
Syndrome, and was initially managed with surgery to re-
pair the oesophageal defect. However, he subsequently
re-leaked, which again was managed surgically with a re-
pair over a T-tube, but then developed a stricture at the
site of injury, which was treated with an oesophageal
stent. This eroded into the airway at its proximal extent,
causing a fistula at 24 cm, with subsequent referral to
our unit (Fig. 4).
After the diagnosis of ADF, three patients underwent
further endotherapy in an attempt to manage the fistula
prior to referral to our unit. Patient 1 had 3 oesophageal
stents placed, but given the proximal location of the
ADF, these all slipped distally. Patient 3, who had devel-
oped an ADF between the airway and a gastric conduit,
most likely after a sub-clinical leak, had endoclips placed
via flexible gastroscopy which failed to close the ADF.
Patient 6 too had a stent
s
placed to treat the fistula with-
out the desired effect.
Surgical intervention for ADF treatment
Three patients (3, 4, 8) underwent a primary repair of
their ADF. Patient 3 required a thoracotomy given that
the ADF was communicating with a gastric conduit and
Patients 4 and 8 had proximally-sited fistulae
approached through the neck. For the former case, an
intercostal flap was interposed between the suture lines
and for the latter two, the strap muscles were similarly
utilised. These fistulae were small and the quality of tis-
sue was sufficiently good to enable primary repair. Pa-
tient 4 presented as an acute emergency following
aspiration -intubated- and a laparotomy was performed
prior to repair, in order to place a feeding jejunostomy
and venting gastrostomy (Table 3).
Fig. 1 CT scan of Patient 1 demonstrating the aerodigestive fistula (arrows)
Table 1 Preceding history and intervention, prior to the diagnosis of ADF
Patient Age at ADF
Diagnosis (years)
Sex Preceding Diagnosis Preceding
Intervention
Chemo-Radiotherapy Preceding Treatment
Related Complication
Previous
Endotherapy
1 60 F SSC
a
Proximal Oesophagus Definitive
chemo-radiotherapy
Chemo-radiotherapy Radiotherapy
related stricture
-×2 stents
-×3 dilations
257 MTB
b
Medical therapy –– –
3 59 M Adenocarcinoma Distal
Oesophagus
Ivor-Lewis
Oesophagectomy
Neo-adjuvant
chemotherapy
Anastomotic leak –
4 73 M Unknown –– – –
532 FTB
b
Medical Therapy –– –
6 33 M Boerhaave’s Syndrome Repair of leak –Re-leak; stricture -× 1 stent
7 55 F Oesophageal B Cell
lymphoma
Chemotherapy Chemotherapy ––
8 29 F Unknown –– – –
a
SCC- Squamous Cell Carcinoma;
b
TB- Tuberculosis
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Two patients (1 and 6) underwent an oesophageal ex-
clusion operation. These were both performed as staged
procedures with delayed reconstruction. The reason for
performing this operation was that in both patients there
was sufficient concern regarding the state of tissue.
Patient 1 had previous radiotherapy for an oesophageal
SCC, on a background of achalasia requiring a myotomy
via thoracotomy several years previously. The degree of
tissue inflammation, scarring and adhesions precluded a
transthoracic resection. Thus, through a neck incision,
the oesophagus was transected above the fistula and an
oesophagostomy fashioned with a synchronous repair of
the fistula. Distally, the oesophagus and OGJ was trans-
ected and a venting gastrostomy fashioned. After a
period of optimisation and treatment of longstanding re-
spiratory disease, the patient underwent reconstruction
utilising a gastric conduit through the retrosternal space.
Patient 6 too had severe inflammation and adhesions in
the chest following his surgical management of Boer-
haave’s syndrome. Exclusion and subsequent reconstruc-
tion with a colonic conduit (he had previously
undergone a distal gastrectomy for benign ulcer disease)
was performed. In both cases, the native tissues were
poor enough to carry a high risk of leak with primary
anastomosis at index surgery.
Patient 2 had a distal ADF, approached through a thora-
cotomy. The right bronchus intermedius was involved,
and the ADF and affected parenchyma was excised as a
segmentectomy, with oesophageal repair. Patient 5 under-
went an exploration of the fistula through a thoractomy
with a lobectomy. The fistula, along with associated nec-
rotic parenchyma was excised en bloc,withasubsequent
suture repair of the oesophagus. Patient 7, after chemo-
therapy for lymphoma, underwent an oesophageal resec-
tion. Again, severe residual inflammation was noted at the
time of the surgery precluding repair of a small (5 mm)
fistula. In addition, our oncology colleagues felt there was
Table 2 Anatomical and Clinical Features of the ADFs
Patient Time to ADF
Development (months)
Fistula Site Fistula Size
(mm)
Main Symptoms Endotherapy to
Treat ADF
1 15 Proximal trachea
20 cm
12 Aspiration ×3 stents
2 > 30 years Oesophagus-right bronchus intermedius
30 cm
12 Recurrent chest infections –
3 21 Gastric conduit-lung 25 cm 16 Recurrent chest infections Endoclip
4 > 30 years Proximal trachea 17 cm 5 Aspiration; Respiratory
embarrassment
–
5 144 Distal oesophagus-lung 38 cm 15 Haemoptysis –
6 3 Oesophagus-carina 24 cm 15 Recurrent chest infections ×3 stents
7 3 Oesophagus- left main bronchus 26 cm 5 Recurrent chest infections –
8 > 20 years Proximal trachea 17 cm 3 Recurrent chest infections –
Fig. 2 Residual ADF (arrow) following treatment for
oesophageal lymphoma
Fig. 3 ADF (arrow) in a proximal location, as seen
by oesophagoscopy
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a reasonable risk of future recurrence of lymphoma, thus
a resection was advocated. A primary repair of the left
main bronchus was performed, utilising a flap muscle
graft. A single-phase operation was performed as the
anastomosis was at a distinct site to the fistula.
Morbidity and mortality
Three patients suffered from complications following sur-
gery. Patient 2 developed a severe respiratory infection on
Day 4 following surgery, requiring bronchoscopic wash-
out. Patient 7 suffered a small anastomotic leak, necessi-
tating prolonged nil oral alimentation. Her nutrition was
maintained with jejunal feeding, and a contrast swallow at
7 weeks confirmed complete healing of the leak, after
which gradual oral feeding was commenced.
Patient 4, who presented with respiratory failure, was af-
fected by severe post-operative recurrent chest infections,
and an inability to wean off mechanical ventilation. This
had been anticipated, and hence a tracheostomy had been
placed at the time of surgery. Despite 4 months of inten-
sive management, he passed away with respiratory failure
and multiple organ dysfunction. He represents the only
mortality in this series.
Normal oral alimentation following surgery was
achieved in all patients, bar patient 4. The time to attain
this milestone ranged from 2 to 3 weeks in all cases,
with the exception of patient 7 who had suffered a leak.
She required supplemental enteral feeding at home for a
short period. We reserve the use of contrast studies and
formal swallow assessment for the very proximal fistulae,
where the risk of leak and aspiration is highest. At the
time of last follow-up, no patient demonstrated clinical
evidence of recurrent ADF.
Hospital episodes statistics (HES)
We performed a search of the English national HES
database to assess the reported incidence of ADFs in the
UK between 2000 and 2012 (Table 4). Only 71 cases
were found. However, we noted that the terms used in
the HES system to record an episode or event related to
an ADF were difficult to identify and, we suspect, many
patients with an ADF were not coded correctly and thus
not recorded. Of the 71 cases, 17 (23.9%) underwent
documented treatment: 9 (12.7%) were treated surgically
and 8 (11.3%) underwent oesophageal stent placement.
Most patients (56.4%) presented with respiratory
symptoms. The 30-day and 90-day mortality rates
were 32.4 and 42.3% respectively, although for cases
Fig. 4 A bronchoscopic image of the ADF (arrow) close to
the carina
Table 3 Surgical Intervention for the Correction of ADF, and Post-Operative Complications
Patient Operation Incision Phases Reconstruction Complications
1 Oesophageal Exclusion and fistula repair
with strap muscle
Left collar
Right PL
a
thoracotomy
Laparotomy
2 phase Retrosternal Gastric conduit –
2 Fistula Repair with intercostal muscle Right PL
a
thoracotomy 1 phase n/a Respiratory infection
3 Fistula Repair with intercostal muscle Right PL
a
thoracotomy 1 phase n/a –
4 Fistula Repair with strap muscle Bilateral collar
Laparotomy
1 phase n/a Respiratory failure
RIP
5 Right lower lobe resection and fistula repair Right PL
a
mini-thoracotomy 1 phase n/a –
6 Oesophageal Exclusion and fistula repair Left collar
Right PL
a
thoracotomy
Laparotomy
2 phase Retrosternal Colonic conduit –
7 Oesophagectomy and fistula repair with
intercostal muscle
Left collar
Right PL
a
thoracotomy
Laparotomy
1 phase Retrosternal Gastric conduit Anastomotic leak
8 Fistula Repair with strap muscle Bilateral collar 1 phase n/a –
a
PL- Postero-lateral
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managed in high-volumes centres this fell to 25 and
31.3% respectively.
Discussion
This series demonstrates the techniques, challenges and
strategies utilised in the surgical management of ADFs.
The key aspect in the approach to such intervention is
the multi-disciplinary nature of care, utilising the experi-
ence of several distinct surgical specialities.
Pre-operative respiratory optimisation should be aggres-
sive, and ideally patients should be weaned off artificial
ventilation prior to surgery [2,3]. As the HES data dem-
onstrates, apart from being rare, there is a deficiency in
accurate diagnosis, coding and documentation of this
condition.
The range of operations that can be utilised reflect the
nature of underlying disease [1–4]. The determinants of
which operation will be performed are mainly the site
and size of the ADF, and the state of the affected tissue-
itself a reflection of the preceding disease and treatment.
For the most proximal ADFs, an approach through a
neck incision is the most desirable. A pivotal stratagem
here involves mobilising the oesophagus circumferen-
tially. This manoeuvre allows the more distal structures
to be brought superiorly into the wound, making further
surgery easier and away from the rigid confines of the
thoracic inlet. If necessary, the medial clavicle and ster-
noclavicular joint can be excised, a procedure which
does not cause future disability [7]. Distal ADFs necessi-
tate a thoracotomy. These ADFs can cause significant
damage to the lung parenchyma, affecting the compli-
ance by causing fibrosis [8]. Thus, where necessary, we
advocate a lobar or segmental resection, with closure of
the associated distal bronchi. Where an oesophagectomy
is indicated, we favour leaving a cuff of oesophageal tis-
sue around the trachea. This enables a dissection plane
away from the airway, and the remnant tissue can be in-
corporated into the repair. This manoeuvre also lessens
the future risk of tracheal stenosis [3].
Although not universally favoured, we have found the
oesophageal exclusion operation a beneficial option in
specific patients. Gross contamination of the airway, or
in patients who have had a previous leak or radiother-
apy, results in significant inflammation and adhesions in
the thorax. Attempting a resection in this circumstance
is hazardous, and if there is no definite indication to
resect, the oesophagus can be safely left in situ. It is im-
portant that a generous venting gastrostomy is sited,
from where the oesophagus can be accessed. We have
successfully performed a retrograde endoscopy through
this, and administered therapeutic agents required for
treatment of disuse oesophagitis. Most importantly, it
enables venting of oesophageal mucous. Some authors
favour a single operation rather than a staged approach
for resection or repair and reconstruction [3,7]. In our
experience, fashioning an anastomosis in a contaminated
field increases the chance of a leak. In such an event, the
fistula has a high chance of recurrence. Most import-
antly, however, a significant leak may necessitate a far
more morbid operative intervention. Indeed, any result-
ant fistula is likely to be more difficult to treat, if this
remains at all possible.
Several other series also demonstrate the complexity
of surgical intervention [1–4,9]. A feature of these re-
ports is that experience is limited to a few specialist
units where an expertise in ADF management is present.
This results in better outcomes, and facilitates an envir-
onment where management is continually improved. In
our series, there was one post-operative death. The
remaining patients all achieved normal alimentation
soon after surgery, with no evidence of ADF recurrence
at a median of 32 months. These results are comparable
to other dedicated units. Mathisen et al demonstrated a
mortality rate of 10.5% in a series of 38 patients, many
of whom underwent tracheal reconstruction, with excel-
lent long-term outcomes [3]. In a subsequent report,
highlighting 35 years’experience in the management of
ADFs, the operative mortality rate fell to 2.8%, reflecting
the effect of concentrating cases in specialist units [1].
Table 4 HES data search for ADFs between 2000 and 2012
(
a
denotes hospitals that perform ≥20 oesophageal cancer
resections per year)
HES data (2000–2012) n%p
Age ≥70 years 35 49.3
Sex
Female 35 49.3
Male 36 50.7
Treatment 17 24
Surgery 9 12.7
Stenting of oesophagus 8 11.3
Unknown 54 76
Presenting Clinical Feature
Pneumonia 18 25.4
Pleural effusion 22 31
Pulmonary embolus 1 1.4
Ischaemic cardiac event 1 1.4
Unknown 29 40.8
All Hospitals
30-day mortality 23 32.4
90-day mortality 30 42.3
Specialist Centres
a
16 22.5
30-day mortality 4 25 0.473
90-day mortality 5 31.3 0.311
Qureshi et al. Journal of Cardiothoracic Surgery (2018) 13:113 Page 7 of 8
Content courtesy of Springer Nature, terms of use apply. Rights reserved.

Shen et al similarly report a low mortality rate of 5.7%,
with a post-operative complication rate of 54.3%, and an
oesophageal leak rate of 11.4% [9]. Baisi et al reported
on 31 patients, of whom 26 underwent simple closure of
the oesophageal and tracheal defects. Operative morta-
lity was 3.2%, with a recurrence rate of 6.4% [10].
Non-operative techniques can be used to manage
ADFs. Oesophageal stenting is the most common inter-
vention, and although it plays a key role in some pa-
tients, they can themselves cause fistulae and may affect
future surgical intervention [11,12]. Newer endoscopic
techniques utilising endoscopic suturing, clip placement
or tissue glue, may have an increasing role in the
management of ADFs in the future. Thus, early referral
to a dedicated unit is advocated, as the whole manage-
ment of the patient can be pursued where the complete
skill-set, including access to novel treatments, is present.
By focusing care in specific units, the expertise in all
facets of management and outcomes can continually
improve.
Conclusion
In summary, for a select group of patients an operative ap-
proach can be a truly life-saving intervention. Although
surgery is not without risk, it offers the best chance of
cure of ADF with a very low risk of recurrence and a
return to normal oral alimentation.
Abbreviations
ADF: Aerodigestive Fistula; HES: Hospital Episodes Statistics;
MDT: Multidisciplinary Team; OGJ: Oesophago-Gastric Junction;
TB: Tuberculosis; TOF: Tracheo-Oesophageal Fistula
Acknowledgements
N/A
Funding
No funding to declare
Availability of data and materials
Made available through UCL research depository (open access)
Authors’contributions
Each author has participated sufficiently in the work to take public
responsibility for this manuscript as per the guidelines of the International
Committee of Medical Journal Editors (ICMJE) criteria. YQ and MM designed
the study, acquired and analysed the data and drafted the manuscript; SM
collected HES data and assisted in writing the manuscript; BM, JG, MH and
DL assisted in interpretation of the data and critically revising the intellectual
content. All authors approved the final version of the manuscript and are
accountable for all aspects of accuracy and integrity related to this work.
Ethics approval and consent to participate
Ethical approval was granted for anonymised patient data under University
College London Hospital research ethics policy.
Consent for publication
Not required
Competing interests
The authors declare that they have no competing interests.
Publisher’sNote
Springer Nature remains neutral with regard to jurisdictional claims in
published maps and institutional affiliations.
Author details
1
Department of Oesophago-Gastric Surgery, University College London
Hospital, 250 Euston Road, London NW1 2BU, UK.
2
Department of Surgery
and Cancer, Imperial College London, London, UK.
3
Department of Thoracic
Medicine, University College London Hospital, London, UK.
4
Department of
Thoracic Surgery, University College London Hospital, London, UK.
Received: 10 April 2018 Accepted: 5 November 2018
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