Congenital bronchoesophageal fistulas in adult patients

doi:10.1016/S0003-4975(95)00326-6

The Annals of Thoracic Surgery

Volume 60, Issue 1, July 1995, Pages 151-155

https://doi.org/10.1016/S0003-4975(95)00326-6 Get rights and content

Background.

Congenital bronchoesophageal fistulas have rarely been reported. Presented here is the 14-year experience of our hospital with this lesion.

Methods.

The study comprises 13 patients; 9 had a Braimbridge type II fistula; 2, a type I fistula; and 2, a type IV fistula. The most frequent site of communication was between the middle esophagus and the right lower lobe of the lung, especially the superior segment. A fistulectomy, with or without pulmonary resection, was performed on each patient.

Results.

All patients had complete relief of symptoms. No operative complications were observed.

Conclusions.

Congenital bronchoesophageal fistulas in adults are usually diagnosed by an esophagography. Symptoms are often nonspecific, and the possibility of a congenital bronchoesophageal fistula should be considered in patients who complain of long-standing unexplainable respiratory symptoms such as coughing and frequent pulmonary infections. The surgical intervention is relatively simple. In many cases, a fistulectomy with simple closure of the openings in both the esophagus and the bronchus is all that is required. Pulmonary resection is needed in some patients with severe bronchiectasis and recurrent pneumonitis.

References (17)

RisherWH et al.
Congenital bronchoesophageal fistula
Ann Thorac Surg
(1990)
HendryP et al.
Benign bronchoesophageal fistulas
J Thorac Cardiovasc Surg
(1985)
BekoeS et al.
Congenital bronchoesophageal fistula in the adult
Chest
(1974)
BeckerRM et al.
Congenital esophagobronchial fistula in a 62-year-old woman
Chest
(1976)
WeissbergD et al.
Bronchoesophageal fistula in adults: congenital or acquired?
J Thorac Cardiovasc Surg
(1990)
SmithDC
A congenital broncho-esophageal fistula presenting in adult life without pulmonary infection
Br J Surg
(1970)
NishimuraY et al.
Congenital esophagobronchial fistula in adult
Nippon Kyobu Geka Gakkai Zasshi
(1987)
KurashigeM et al.
Congenital esophagobronchial fistula in a 39-year-old male
Nippon Kyobu Geka Gakkai Zasshi
(1985)

There are more references available in the full text version of this article.

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Two unusual cases of adult onset congenital bronchoesophageal fistulas treated with Fistula division
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Symptoms are nonspecific, bouts of cough when swallowing liquids (Ohno’s sign) is considered pathognomonic and is present in 65% of all cases. The duration of symptoms before diagnosis varies from 6 months to 50 years [4]. Both patients had been tolerant to their symptoms for many years.
Adult onset congenital bronchoesophageal fistula is a very rare entity. We report 2 cases of adult onset type II congenital bronchoesophageal fistula between the distal thoracic esophagus and the lower lobe superior segmental bronchi surgically treated through a right and left thoracotomy, respectively. In both cases the fistula was transected and sutured with no parenchyma resection. Both patients had an uneventful recovery. Resection of the underlying parenchyma during surgery for bronchoesophageal fistula is not always necessary as the lung can heal in time after performing just fistulectomy.
Tracheobronchial and pulmonary parenchymatous congenital abnormalities requiring surgical treatment in adults
2012, Revue de Pneumologie Clinique
La plupart des malformations trachéobronchiques et parenchymateuses pulmonaires sont découvertes dans les premiers mois de la vie, mais certaines peuvent n’être diagnostiquées qu’à l’âge adulte. Sont concernés l’arbre trachéobronchique (sténose trachéale et/ou bronchique, kyste bronchogénique, atrésie bronchique, fistule œsotrachéale, fistule œsobronchique, et diverticule trachéal) et le parenchyme pulmonaire (séquestration, malformation adénoïde kystique, emphysème lobaire, hypoplasie lobaire ou pulmonaire). Une prise en charge chirurgicale est parfois nécessaire, et ces cas rares doivent être connus afin de ne pas négliger une malformation potentiellement dangereuse.
Most tracheobronchial and parenchymatous congenital abnormalities of the respiratory system are diagnosed in early life. However, some lesions may be initially silent and diagnosed only in adulthood. These cases included congenital abnormalies of the tracheobronchial tract (tracheal and/or bronchial stenosis, bronchogenic cysts, bronchial atresia, oesotracheal fistula, oesobronchial fistula, and tracheal diverticulum), and lung parenchyma itself (pulmonary sequestration, congenital cystic adenomatoïd malformation, lobar emphysema, lobar or lung hypoplasia). To avoid dreadful complications, these rare cases deserve surgical management, and must be known by chest physicians and surgeons.
Long-term results of surgical treatment in benign bronchoesophageal fistula
2007, Journal of Thoracic and Cardiovascular Surgery
Citation Excerpt :
We had 2 cases of malignant BEFs managed surgically at our institution from 1995 to 2005, and they were excluded from our study. However, BEFs could also develop from benign etiologies such as infection, trauma, and esophageal diverticulum, although they have been rarely reported so far1,3-9 Since Heiderich10 first demonstrated a fistula between the right main stem bronchus and the esophagus in 1916, only about 120 cases of benign BEF have been published in the world literature.1 Even the largest single institutional series reported by Braimbridge and Keith11 in 1965 included 3 cases from their own experience and 20 cases that had been released in the literature until that time.
Benign bronchoesophageal fistula is a rare complication resulting from various diseases. The objectives of the study are to report our surgical experience in patients with benign bronchoesophageal fistulas and to evaluate the long-term results of surgical management.
Between 1995 and 2005, a total of 14 patients (mean age, 52.5 years; male/female = 6:8) underwent operations for benign bronchoesophageal fistulas. The etiology of the fistulas included traction esophageal diverticula in 7 patients and tuberculous lymphadenopathy in 6. Primary repair of the fistula was performed in all patients and a concomitant lobectomy in 6.
There was no in-hospital mortality. Postoperative complications occurred in 1 patient (empyema resulting from a leakage at the repair site). Postoperative esophagography revealed a leakage at the repair site in 1 patient. There was 1 late death with a mean follow-up of 43.9 months. The cause of death was aspiration pneumonia resulting from recurrent fistula. Two of 8 patients who did not undergo a lobectomy had persistent bronchiectasis, whereas none of those who underwent a concomitant lobectomy had any recurrent pneumonia or bronchiectasis. The overall survival was 100% at 5 years and the mean survival time was 124 months (95% confidence interval, 106.5-142.2 months).
We conclude from these data that early detection and definitive surgical repair of benign bronchoesophageal fistulas can yield high success rates with excellent early outcomes and good long-term results.
Congenital esophagobronchial fistula discovered in an adult
2006, Revue de Pneumologie Clinique
Les fistules œso-bronchiques congénitales sont des anomalies rares, découvertes en période néo-natale car leur symptomatologie est bruyante. A l’inverse, si elles ne sont découvertes qu’à l’âge adulte, c’est que leur symptomatologie est beaucoup plus fruste, limitée le plus souvent à une toux lors d’ingestion de liquides, voir absente. Moins de 200 cas sont rapportés dans la littérature.
Les auteurs rapportent un nouveau cas de fistule œso-bronchique congénitale type II : une patiente de 59 ans, hospitalisée pour bilan d’une toux lors d’ingestion de liquide. Le bilan radiologique a retrouvé une dilatation des bronches basales gauches et suspecté une fistule œsobronchique. Le transit œsogastro-duodénal a mis en évidence une fistule entre le tiers moyen de l’œsophage et les bronches du segment postéro-basal gauche. Une fistulectomie et une lobectomie du lobe inférieur gauche ont été réalisées, avec une bonne évolution. Les auteurs insistent sur l’intérêt de rechercher ce diagnostic devant une dilatation des bronches localisée, chez un patient sans antécédents et présentant une toux synchrone à l’ingestion de liquide.
Congenital esophagobronchial fistulae are uncommon anomalies generally discovered during the neonatal period due to overt symptoms. Rarely symptoms are discrete, leading to discovery in adulthood; exceptionally the patients are totally symptom free or present minimal signs such as coughing at ingestion of fluids. Less than 200 adult cases have been reported in the literature.
We report a new case of this type II congenital esophagobronchial fistula. A 59-year-old woman was hospitalized for exploration of cough at ingestion of fluids. The radiographic work-up reveal dilatation of the left bronchial tree and suspected esophagobronchial fistula. An esophagogastroduodenal barium study demonstrated a fistula between the mid esophagus and the left posterobasal bronchus. Fistulectomy and left lower lobectomy were performed. Outcome was favorable. The diagnosis of adult esophagobronchial fistula should be entertained in patients with an uneventful history who present localized bronchial dilatation associated with cough induced by fluid ingestion.
Congenital bronchoesophageal fistula in the adult: A case report
2000, American Journal of Gastroenterology
Citation Excerpt :
The youngest reported was in a 9-day-old boy and the oldest in an 83-yr-old man, with the most common location being between the right lower lobe and middle third of the esophagus (1). Fewer cases have been reported that communicate with the left lung, and there are only two cases reported in the literature of a fistula between the esophagus and left main bronchus (2, 3). Symptoms may begin at any time and are often intermittent.
Congenital bronchoesophageal fistula is a rare anomaly that may go undiagnosed for many years. There are <200 reported cases in the literature. Patients usually present with chronic cough, a history of multiple pneumonias, and, less often, hemoptysis. The most sensitive test to diagnose a bronchoesophageal fistula is barium esophagogram. Surgical resection is the treatment of choice and is definitive in almost all cases. We present the case of a 52-yr-old woman with a history of cough and multiple pneumonias who sought medical treatment for abdominal pain. We discuss the presentation and the radiographic, endoscopic, and surgical findings, along with a review of the literature.

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: This study is supported by grant 01-94-056 from the Seoul National University Hospital Research Fund.

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Original articleCongenital bronchoesophageal fistulas in adult patients

Background.

Methods.

Results.

Conclusions.

Ann Thorac Surg

J Thorac Cardiovasc Surg

Chest

Chest

J Thorac Cardiovasc Surg

A congenital broncho-esophageal fistula presenting in adult life without pulmonary infection

Br J Surg

Congenital esophagobronchial fistula in adult

Nippon Kyobu Geka Gakkai Zasshi

Congenital esophagobronchial fistula in a 39-year-old male

Nippon Kyobu Geka Gakkai Zasshi

Original article
Congenital bronchoesophageal fistulas in adult patients