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CASE REPORT
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A rare case of congenital esophagobronchial fistula in an
adult
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V.YaminiChitra, K. N. Paramesh, Alamelu Haran, Nitin D. Tengli
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AC
V.YaminiChitra1, K. N. Paramesh2, Alamelu Haran3, Nitin D.
Tengli4
Affiliations: 1MS, (General Surgery), DNB, MCH (Surgical
Gastroenterology), Associate Professor, Dept of Surgical
Gastroenterology and Bariatric centre, Vydehi Institute
of Medical Sciences and Research Centre, Bangalore,
Karnataka, India; 2MS, (General Surgery), (DNB Surgical
Gastroenterology), Senior Resident, Dept of Surgical
Gastroenterology and Bariatric Centre, Vydehi Institute
of Medical Sciences and Research Centre, Bangalore,
Karnataka, India; 3MD (Tuberculosis and Chest Diseases),
Professor and HOD, Department of Pulmonary Medicine
Vydehi Institute of Medical Sciences and Research Centre,
Bangalore, Karnataka, India; 4MS, (General Surgery) Senior
Resident, Department of Surgical Gastroenterology and
Bariatric Centre, Vydehi Institute of Medical Sciences and
Research Centre, Bangalore, Karnataka, India
Corresponding Author: V.YaminiChitra, Associate Professor,
Department of Surgical Gastroenterology and Bariatric
Centre, Vydehi Institute of Medical Sciences and Research
Centre, Bangalore, Karnataka, India. #82, Nallurahalli,
Whitefield, Bangalore, Karnataka, India, Postal Code
560 066; Tel: +919611108242, Fax: 08028412956;
Email: [email protected]
Received: 28 October 2014
Accepted: 27 November 2014
Published: 01 February 2015
Abstract
Introduction: Congenital esophagobronchial
fistulas in adults are extremely rare, acquired
fistulas being more common. The aim of this
paper is to present a rare case of congenital
esophagobronchial fistula in a 37-year-old male
of type II Braimbridge’s classification and to
emphasize on the diagnostic modality of choice
and the appropriate mode of treatment. Case
Report: A 37-year-old male presented with
chronic cough with ingestion of food, especially
liquids of 13 years duration and recent onset
hemoptysis. He was evaluated with upper
gastrointestinal
endoscopy,
bronchoscopy,
computed tomography scan of chest and the
definitive test was barium swallow which
confirmed it.
He underwent transthoracic excision of the
fistula with repair of both esophageal and
bronchial ends. A peroperative endoscopy
helped localization of the tract. Postoperative
outcome was excellent with no leak and patient
is totally asymptomatic after 12 weeks of surgery.
Congenital
esophagobronchial
Conclusion:
fistulas in adults, due to insidious nature need
high index of suspicion as early diagnosis by
barium swallow and surgical treatment gives
excellent results. Peroperative endoscopy is
mandatory to localize the tracts, helps do an
intraoperative leak test and avoid esophageal
stenosis during repair.
Keywords: Barium swallow, Congenital esophagobronchial fistula, Peroperative endoscopy, Transthoracic excision
How to cite this article
YaminiChitra V, Paramesh KN, Haran A, Tengli ND.
A rare case of congenital esophagobronchial fistula
in an adult. Int J Case Rep Images 2015;6(2):*****.
International Journal of Case Reports and Images, Vol. 6 No. 2, February 2015. ISSN – [0976-3198]
Int J Case Rep Images 2015;6(2):**–**.
www.ijcasereportsandimages.com
doi:10.5348/ijcri-201513-CR-10474
INTRODUCTION
2
Patient underwent right posterolateral thoracotomy.
There were minimal adhesions around the fistulous site
which was identified about 5 cm below the level of entry
of azygos vein into superior vena cava. Azygos vein was
isolated, ligated and cut to aid esophagus to be encircled,
to localize the fistulous tract. As the guide wire was not
palpable through the tract, intraoperative endoscopy
was done and fistula tract location was confirmed. The
fistulous tract was dissected, it was 10 mm long. The
tract was excised, both the esophageal and bronchial
ends were healthy and were closed with 4-0 Vicryl. An
intraoperative leak test was done using endoscopy which
confirmed the integrity of the repair and an intercostal
drain placed.
Postoperative period was uneventful. Intercostal
drain was removed on postoperative day-4 after a barium
swallow to confirm that there was no leak and patient was
discharged on postoperative day-6, after starting oral soft
diet.
Histopathology showed the mucosa to be lined by
stratified squamous epithelium. There was no evidence
of inflammation, granuloma or carcinoma confirming
the congenital nature of the fistula. Patient is totally
asymptomatic after 12 weeks of surgery.
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Fistula between esophagus and bronchus may
be congenital or acquired. Acquired fistulas can be
inflammatory, traumatic or neoplastic. Congenital
esophago bronchial fistula (EBF) with atresia present
immediately in infancy, are sudden and are diagnosed
early and treated [1]. Congenital EBF without atresia are
insidious, can present in adulthood also or missed often
especially if they communicate with a lobar bronchus [1].
To differentiate between congenital and acquired EBF
in an adult is usually difficult [2]. Adult EBF may present
acutely with sudden respiratory distress, chronically
with repeated respiratory infections or can be totally
asymptomatic. Less than 200 cases of adult EBF are
reported so far in literature [2].
Herein, we report a rare case of 37-year-old male
with congenital EBF without atresia (Type 2 according to
Braimbridge’s classification) [1] which was diagnosed by
barium esophagogram. Patient underwent transthoracic
fistulectomy with the repair of both esophageal and
bronchial ends. Postoperative outcome was successful
with complete resolution of symptoms and closure of
fistula.
Chitra et al. CASE REPORT
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A 37-year-old male presented to our department
with history of cough with expectoration immediately
after taking food, especially liquids for last 13 years. He
had two bouts of hemoptysis in the last 15 days which
had made him seek medical attention. Clinically, chest
on auscultation had crackles in right infra-scapular
region. Chest X-ray revealed patchy opacities in the right
lower zone paracardiac region. Computed tomography
(CT) scan of thorax revealed consolidation in posterior
segment of the right lower lobe with no evidence of lung
sequestration or cyst. Upper gastrointestinal endoscopy
showed a fistulous opening in the mid esophagus 32 cm
from the incisor teeth. It did not show any evidence of
malignancy, granulomatous disease or any other acquired
basis for the fistula. Simultaneously, methylene blue was
injected into the fistulous tract and bronchoscopy was
done which was normal. Barium swallow showed fistulous
communication between mid oesophagus and right lower
lobe bronchus at lower border of T7 with barium passing
downward into the right lung.
Preoperative evaluation done included pulmonary
function tests, echocardiogram and electrocardiogram.
Incentive spirometry was started for better postoperative
outcome. Preoperatively, 1 fr size guide wire was
introduced endoscopically into the fistulous tract to aid
identification of fistula.
Figure 1: Barium esophagogram showing esophagobronchial
fistula between the middle third of the esophagus and the right
lower bronchus with downward passage of barium (shown with
arrow).
International Journal of Case Reports and Images, Vol. 6 No. 2, February 2015. ISSN – [0976-3198]
Int J Case Rep Images 2015;6(2):**–**.
www.ijcasereportsandimages.com
Chitra et al. 3
DISCUSSION
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The majority of EBF in adults are acquired. The usual
causes are inflammatory like tuberculosis, trauma and
neoplasms. EBF was first described as early as in 1916 by
Heiderich [3]. Congenital EBF are rare, three times more
common on the right than the left [4].
According to Braimbridge’s classification, type I
is a fistula associated with a wide-necked congenital
diverticulum of the esophagus with inflammation at the
tip. Type II, which is the simplest and most common,
consists of a short tract running directly from the
Figure 4: Intraoperative photograph with intraoperative upper
gastrointestinal endoscopy (light of endoscopy is visible through
the tract).
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Figure 2: Endoscopic findings: The fistula opening in the middle
esophagus (arrow).
Figure 3: Intraoperative photograph showing bronchoesophageal
fistula. The fistula opening in middle esophagus (shown with
arrow), proximal, distal esophagus and fistulous tract is also
looped.
Figure 5: Postoperative barium esophagogram showing normal
esophagus and no evidence of fistula.
esophagus to the lobar or segmental bronchus. In type
III, the fistulous tract connects the esophagus to a cystic
pulmonary change, and in type IV a fistula runs into a
sequestered pulmonary segment. The patient described
here is of type 2 according to this classification [1].
Criteria for congenital EBF are suggested pathologically
by the absence of surrounding inflammation and adherent
lymph nodes along with the presence of a mucosa and a
definitive muscularis mucosa within the fistulous tract.
Surgically, uncomplicated and easy dissection of the
fistula and absence of inflammation suggests a congenital
fistula [5].
Reasons for delay in the onset of symptoms and
presentation in the adult may be due to
International Journal of Case Reports and Images, Vol. 6 No. 2, February 2015. ISSN – [0976-3198]
Int J Case Rep Images 2015;6(2):**–**.
www.ijcasereportsandimages.com
4
interposition when needed, gives permanent cure and
relief of symptoms in the patients with EBF.
CONCLUSION
Congenital esophagobronchial fistulas in adults,
due to rare occurrence and insidious nature need high
index of suspicion as early diagnosis by barium swallow
and surgical treatment by open or minimally invasive
technique gives excellent results.
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Author Contributions
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(i) A fistula tract which runs upwards and may close
during swallowing. But in our patient barium was passing
downward during the swallow.
(ii) A membrane which later ruptures.
(iii) A fold of esophageal mucosa which overlaps the
orifice but subsequently becomes less mobile [1].
Both sexes are affected almost equally (male 53 % and
females 47 %) [6].
Symptoms are often intermittent, due to chronic
bronchopulmonary infection. They are chronic cough
(96%), pneumonia (56%), hemoptysis (17%). Ohno’s
sign characterized by symptoms of strangulation and
paroxysmal coughing with swallowing liquids occurs in
the presence of a very large communication [7].
The diagnosis is usually made by barium esophagogram
[1]. A thin barium is given, in a position where patient
gets most of the symptoms. Endoscopy and bronchoscopy
should be done but will not always demonstrate the
opening in esophagus and bronchus [4]. Ramo et al. [4]
have shown that bronchoscopy is negative in 67% of cases
and esophagoscopy in 40%. Bronchoscopy can be normal,
if the communication is in the distal segmental portion of
the tract as in our case [7]. Dynamic bronchoscopy with
pediatric bronchoscope may help [7]. Right bronchus
is most commonly affected [7]. Computed tomography
scan may help to rule out type 4 fistulas and aortography
can document the sequestrated lung [8] and aid in
pulmonary resection if needed. Peroperative endoscopy
helps accurately in identifying the tracts [2], to confirm
patency of the esophagus if the repair is very proximal or
on the esophageal wall itself, as in wide short tracts and
also aids in doing an intraoperative leak test as done in
our case.
Thoracotomy and resection of the fistulous tract with
primary repair of both the bronchial and esophageal
defects with 4-0 Vicryl the ideal treatment of choice.
Interposition of pleural/diaphragmatic flap helps to
reduce the recurrence. In our case, it was not done as the
tract was long and the bronchial and esophageal tissue
post repair were healthy. The diseased lung tissue and
sequestrated lobes if any should be resected at the same
time.
Postoperative barium swallow helps document the
healed defect in the esophagus.
Endoscopic management by submucosal dissection
and isolation of the fistulous tract with clipping has
been tried in tracheoesophageal fistula but has been
unsuccessful [9]. Other endoscopic techniques like
histoacryl glue injection have been tried in recurrent
congenital trachea esophageal fistulas, or when patient
has refused surgery with varied results.
Thoracoscopic repair assisted by peroperative
endoscopy and stapling of the fistulous tract by surgeons
experienced in minimally invasive surgery can reduce the
morbidity associated with open thoracotomy [10].
Either thoracoscopic or open thoracotomy repair
with excision of the fistulous tract and good repair
of the esophageal and bronchial defects with tissue
Chitra et al. V. Yamini Chitra – Substantial contributions to
conception and design, Acquisition of data, Analysis
and interpretation of data, Drafting the article, Revising
it critically for important intellectual content, Final
approval of the version to be published
K. N. Paramesh – Substantial contributions to
conception and design, Acquisition of data, Analysis and
interpretation of data, Drafting the article, Final approval
of the version to be published
Alamelu Haran – Substantial contributions to
conception and design, Acquisition of data, Analysis and
interpretation of data, Drafting the article, Final approval
of the version to be published
Nitin Tengli – Substantial contributions to conception and
design, Acquisition of data, Analysis and interpretation of
data, Drafting the article, Final approval of the version to
be published
Guarantor
The corresponding author is the guarantor of submission.
Conflict of Interest
Authors declare no conflict of interest.
Copyright
© 2015 V. Yamini Chitra et al. This article is distributed
under the terms of Creative Commons Attribution
License which permits unrestricted use, distribution
and reproduction in any medium provided the original
author(s) and original publisher are properly credited.
Please see the copyright policy on the journal website for
more information.
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About the Authors
Article citation: YaminiChitra V, Paramesh KN, Haran A, Tengli ND. A rare case of congenital esophagobronchial
fistula in an adult. Int J Case Rep Images 2015;():*****.
V.YaminiChitra is an Associate Professor at Department of Surgical Gastroenterology and Bariatric
centre Vydehi Institute of Medical Sciences and Research Centre, Bangalore, Karnataka, India. She
earned the undergraduate degree Bachelor of Medicene and Bachelor of Surgery from Coimbatore
Medical college, Dr. MGR medical University, India and postgraduate degree Master of Surgery
from Stanley Medical college, Chennai, Dr. MGR medical University, India. She has got Diplomate
in Surgery (DNB) from National Board of Examinations, India and Master of Chirurgery in Surgical
Gastroenterology from Madras Medical College, Dr. MGR medical University, India. Her research
interests include hepatobiliary and pancreatic surgeries, specifically benign biliary strictures, chronic calcific
pancreatitis. Email: [email protected]
AC
K. N. Paramesh is Senior resident, Department of Surgical Gastroenterology and Bariatric centre,
Vydehi Institute of Medical Sciences And Research Centre, Bangalore, Karnataka, India. He earned
the undergraduate degree Bachelor of Medicene and Bachelor of Surgery from Sri Devaraj urs Medical
college and research centre, RGUHS medical University, Kolar, India and postgraduate degree Master
of Surgery from BLDEA,S Shri BM patil medical college and research centre, Bijapur under RGUHS
medical University,Karnataka, India. He has undergone DNB in surgical gastroenterology from Gem
hospipital and reseach centre, Coimbatore, National Board of Examinations, India. He has published:
A Case report titled “Pica — a case of acuphagia or hyalophagia?” Kariholu PL, Jakareddy R, Hemanth Kumar M,
Paramesh KN, Pavankumar Indian J Surg. Jun 2008; 70(3): 144–146. Jul 24, 2008. PMCID: PMC3452446. His
research interests include Advanced laparoscopy in GI cancer, hepatobiliary and pancreatic surgeries and bariatric
surgery. Email: [email protected]
Alamelu Haran is Professor & HOD, Department of Pulmonary medicine Vydehi Institute of Medical
Sciences And Research Centre, Bangalore, Karnataka, India. She earned the undergraduate degree
Bachelor of Medicine and Bachelor of Surgery from Grant medical college, Mumbai. Bombay University,
India ) and postgraduate degree MD (Tuberculosis and Chest Diseases), from Topiwala National Medical
College, Mumbai. Bombay University, India. Her research interests include Pulmonary function tests,
Sleep medicine and Bronchoscopy. She has published original article—“Clinical evaluation of a sublingual vaccine for prevention of exacerbations in COPD” has been accepted for publication in upcoming
issue of Asian Journal of Medical Sciences (AJMS) volume 6 issue 4 (july-aug 2015). Email: [email protected]
com
Nitin D. Tengli is Senior resident, Department of Surgical Gastroenterology and Bariatric centre,
Vydehi Institute of Medical Sciences and Research Centre, Bangalore, Karnataka, India. He earned the
undergraduate degree Bachelor of Medicene and Bachelor of Surgery from Mahadevappa Rampura
Medical college, RGUHS medical University,Gulbarga, India and postgraduate degree Master of
Surgery from Mahadevappa Rampura Medical college, RGUHS medical University, Gulbarga, India.
International Journal of Case Reports and Images, Vol. 6 No. 2, February 2015. ISSN – [0976-3198]