HOME HELP FEEDBACK SUBSCRIPTIONS ARCHIVE SEARCH TABLE OF CONTENTS		QUICK SEARCH:   [advanced] Author: Keyword(s): Year:  Vol:  Page:

This Article Abstract Alert me when this article is cited Alert me if a correction is posted Citation Map Services Email this article to a friend Similar articles in this journal Similar articles in PubMed Alert me to new issues of the journal Add to Personal Folders Download to citation manager Author home page(s): Severi P. Mattila Permission Requests Citing Articles Citing Articles via HighWire Citing Articles via Google Scholar Google Scholar Articles by Rämö, O. J. Articles by Mattila, S. P. Search for Related Content PubMed PubMed Citation Articles by Rämö, O. J. Articles by Mattila, S. P. Related Collections Related Article

Ann Thorac Surg 1995;59:887-890
© 1995 The Society of Thoracic Surgeons

---

Orginal Articles

Congenital Bronchoesophageal Fistula in the Adult

Department of Thoracic and Cardiovascular Surgery, Helsinki University Central Hospital, Helsinki, Finland

Accepted for publication November 5, 1994.

Abstract

Congenital bronchoesophageal fistula is a rare clinical entity in adult patients. This anomaly may cause various symptoms such as respiratory infections, coughing bouts when eating or drinking, and even hemoptysis. We present a report on 9 patients with bronchoesophageal fistulas who were treated in our hospital during the last 30 years. One patient died of pulmonary complications before definitive treatment of the fistula. Other patients recovered from surgical closure of the fistula without complications. Based on our experience, these patients should be treated surgically without delay. Observation may result in fatal pulmonary complications.

See also page 890.

Congenital bronchoesophageal fistula is a rare clinical problem in adult patients. Fistulas between the esophagus and the respiratory system usually are diagnosed during the neonatal period, but in certain cases they may not appear until adult life [1]. Braimbridge and Keith [2] classified congenital bronchoesophageal fistulas into four types. In type I a fistula is associated with a wide-necked congenital diverticulum of the esophagus. Type II, which is the simplest and most common, consists of a short tract running directly from the 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 diagnosis of this condition may be difficult due to its insidious clinical course. If a respiratory--esophageal fistula is not suspected and is therefore left untreated, it may lead to fatal complications despite the benign nature of this anomaly [3]. During the last 30 years we have treated 9 cases of congenital bronchoesophageal fistula in our hospital. We present our experience in treating these cases and a short review of various options for diagnosis and treatment of this anomaly.

Material and Methods

Nine cases of bronchoesophageal fistula have been treated in our department since 1963. Five patients were female and 4 were male. The mean age of the patients was 36 years (range, 17 to 64 years). Patient data are summarized in Table 1. In all cases, the congenital nature (squamous epithelium with mucosa and muscularis musosae) of the fistula was verified by histologic examination.

Fistula between the bronchus/trachea and the esophagus results from failed tracheoesophageal separation in the early stage of embryonic development [4]. Congenital bronchoesophageal fistulas are equally common in male and female patients. However, these fistulas are almost three times as common in the right than in the left side [3]. In the present series the majority (67%) of the fistulas were on the right side. According to the classification by Braimbridge and Keith [2], type II fistula is the most common. In our patients 8 cases presented type II fistula (89%) and only 1 was classified as type IV fistula.

A fistula may cause symptoms in childhood but may not appear until adult life [2–4]. A bronchoesophageal fistula typically is associated with repeated and persistent respiratory infections and coughing bouts when eating. Hemoptysis also is seen occasionally, as in 1 of our patients. Bronchoesophageal fistulas may, however, be silent for several decades due to various reasons. If the fistula runs upward it may cause symptoms only in certain positions, such as lying on either side. A congenital fistulous tract may be sealed by a thin membrane, which may rupture completely or start to act as a one-way valve later in life and produce various symptoms [5, 6]. In our series a 25-year old man had a pulmonary abscess at the age of 3 years and was then virtually symptomless for 20 years. On the other hand, a 64-year old woman (Figs 1, 2) had symptoms for only 3 years before operation; ie, the fistula was silent for 61 years. In elderly patients esophagorespiratory fistula is not always suspected and it may result in fatal complications before definitive treatment, as occurred in 1 of our patients.

View larger version (98K): [in this window] [in a new window]   Fig 1. . Barium esophagogram of a 64-year-old woman who had a congenital bronchoesophageal fistula that had been silent for 61 years. Bronchoesophageal fistula is evident from the esophagus to the right main bronchus. Both esophagoscopy and bronchoscopy also were diagnostic in this case.

View larger version (172K): [in this window] [in a new window]   Fig 2. . Operative photograph of the fistula taken from the same patient as in Figure 1. The fistula (arrow) was excised, both openings were closed with sutures, and the closure was covered with pedicled pleural flaps. Her recovery was uneventful.

Division of the fistula and excision of any permanently damaged lung segment has been considered the method of choice in the treatment of this disease. Parry and Juma [7] recently have treated successfully a congenital bronchoesophageal fistula with the use of local application of sodium hydroxide and acetic acid to both bronchial and esophageal aspects of the fistula. They suggest that endoscopic treatment be used for patients who are too sick to undergo thoracotomy or who decline the operation. However, it can offer only a temporary closure of the fistula, because the cause cannot be confirmed.

Weissberg and Kaufman [8] used a surgical stapler in 2 patients to close the fistula without dividing it. They did not observe recurrence more than a year after the operation and they believed that less trauma and operating time would decrease morbidity and mortality, especially in elderly people. The largest drawback of this method is the lack of histologic diagnosis, and limited space between esophagus and bronchus may result in even more tissue trauma than simple excision and closure of the openings.

Because the etiology of this anomaly is neither infectious nor malignant, bronchial and esophageal openings may be closed directly after excision of the fistula without compromising the healing process. On the other hand, we do not think that excision of the fistula and securing the closure by the aid of pleural flaps would increase morbidity in these patients. In 4 of our patients, pedicled pleural flaps were used as presented by Grillo and Wilkins [9], and 4 patients underwent direct closure of the fistula. All patients recovered equally well without complications. The histopathologic diagnosis must be precise before the treatment of a bronchoesophageal fistula. This is seldom possible without excision of the fistula. Even though it seems tempting to use less invasive techniques than open thoracotomy, it should be noted that endoscopic treatment may overlook other causes, such as inflammatory and malignant fistulas.

In conclusion, our experience shows that these patients should be treated surgically as soon as possible after the diagnosis is established. Delays in treatment may cause pulmonary complications and even fatality.

Footnotes

Address reprint requests to Dr Rämö, Department of Thoracic and Cardiovascular Surgery, Helsinki University Central Hospital, Haartmaninkatu 4, FIN-00290 Helsinki, Finland.

References

1. Azoulay D, Regnard JF, Magdeleinat P, Diamond T, Rojas-Miranda A, Levasseur P. Congenital respiratory-esophageal fistula in the adult. Report of nine cases and review of the literature. J Thorac Cardiovasc Surg 1992;104:381–4.[Abstract]
2. Braimbridge MV, Keith HI. Oesophago-bronchial fistula in the adult. Thorax 1965;20:226–33.[Free Full Text]
3. Risher WH, Arensman RM, Ochsner JL. Congenital bronchoesophageal fistula. Ann Thorac Surg 1990;49:500–5.[Abstract]
4. Smith BD Jr, Mikaelian DO, Cohn HE. Congenital bronchoesophageal fistula in the adult. Ann Otol Rhinol Laryngol 1987;96:65–7.[Medline]
5. Demong CV, Grow JB, Heitzman GC. Congenital tracheoesophageal fistula without atresia of the esophagus. Am Surg 1959;25:156–62.[Medline]
6. Negus VE. Oesophagus from a middle-aged man, showing a congenital opening into the trachea. Proc R Soc Med 1929;22:527.
7. Parry W, Juma A. Congenital bronchoesophageal fistula [Letter]. Ann Thorac Surg 1991;51:346–7.[Medline]
8. Weissberg D, Kaufman M. Bronchoesophageal fistula in adults: congenital or acquired? [Letter]. J Thorac Cardiovasc Surg 1990;99:756–7.[Medline]
9. Grillo HC, Wilkins EW Jr. Esophageal repair following late diagnosis of intrathoracic perforation. Ann Thorac Surg 1975;20:387–99.[Abstract]

This article has been cited by other articles:

				H. K. Kim, Y. S. Choi, K. Kim, J. Kim, and Y. M. Shim Long-term results of surgical treatment in benign bronchoesophageal fistula J. Thorac. Cardiovasc. Surg., August 1, 2007; 134(2): 411 - 414. [Abstract] [Full Text] [PDF]

				S. Griffo, P. Stassano, G. Iannelli, L. Di Tommaso, M. Cicalese, M. Monaco, and G. Ferrante Benign bronchoesophageal fistula: Report of four cases J. Thorac. Cardiovasc. Surg., May 1, 2007; 133(5): 1378 - 1379. [Full Text] [PDF]

				O. J. Ramo, J. A. Salo, J. Isolauri, M. Luostarinen, and S. P. Mattila Tuberculous Fistula of the Esophagus Ann. Thorac. Surg., October 1, 1996; 62(4): 1030 - 1032. [Abstract] [Full Text]

				R. Saldanha, S. V. Srikrishna, L. Talwalkar, K. Kilpadi, M. Vasnaik, O. J. Ramo, J. A. Salo, and S. P. Mattila Diagnosis of Congenital Bronchoesophageal Fistula During Anesthesia Ann. Thorac. Surg., June 1, 1996; 61(6): 1875 - 1876. [Full Text]

This Article Abstract Alert me when this article is cited Alert me if a correction is posted Citation Map Services Email this article to a friend Similar articles in this journal Similar articles in PubMed Alert me to new issues of the journal Add to Personal Folders Download to citation manager Author home page(s): Severi P. Mattila Permission Requests Citing Articles Citing Articles via HighWire Citing Articles via Google Scholar Google Scholar Articles by Rämö, O. J. Articles by Mattila, S. P. Search for Related Content PubMed PubMed Citation Articles by Rämö, O. J. Articles by Mattila, S. P. Related Collections Related Article