| Abstract|| |
Broncho-esophageal fistula (BEF) are quite rare in adults, more so the congenital variety. The common causes of BEF in adults include infections, trauma, and malignancies. We report a rare case of congenital BEF manifesting in adulthood with repeated pulmonary infections. We emphasize mainly on the preoperative preparation and perioperative management of this patient. It is essential to have a high index of suspicion to diagnose congenital BEF in adults. Anesthesiologists play an important role in successful management of these cases.
Keywords: Broncho-esophageal fistulae, One lung ventilation, Thoracic epidural
|How to cite this article:|
Ranjan R V, Ramachandran T R, Veliath DG. Anesthetic management of congenital broncho-esophageal fistula in an adult. Ann Card Anaesth 2012;15:299-301
|How to cite this URL:|
Ranjan R V, Ramachandran T R, Veliath DG. Anesthetic management of congenital broncho-esophageal fistula in an adult. Ann Card Anaesth [serial online] 2012 [cited 2021 Mar 1];15:299-301. Available from: https://www.annals.in/text.asp?2012/15/4/299/101865
| Introduction|| |
Broncho-esophageal fistula (BEF) are quite rare in adults, more so the congenital variety.  Most common causes of BEF in adults are malignancies, trauma, and infections. If not associated with esophageal atresia, congenital varieties are difficult to diagnose during infancy. We report a rare case of congenital BEF manifesting in adulthood with repeated pulmonary infections.
| Case Report|| |
A 45-year-old male presented with cough of 12 years duration and recurrent attacks of lower respiratory tract infection. Cough was more in supine and left lateral decubitus position and aggravated by intake of food. Sputum production per day was around 500 ml and it mainly contained ingested food materials. There was no history of tuberculosis (TB), instrumentation of esophagus and airway, or surgical interventions of thorax. No history of corrosive acid ingestion was noted. The patient was in good general health. On general examination, he had grade 2 clubbing, and on auscultation he had decreased air entry and coarse crepitations on the right basal area of the chest. His electrocardiogram, transthoracic echocardiography, liver function, and renal function tests were within normal limits. The chest X-ray showed pulmonary infiltrates in the middle and inferior zones of the right lung. Barium swallow showed an abnormal communication between the esophagus and the right bronchial tree [Figure 1]. Esophagoscopy [Figure 2] showed the opening of the fistula but there was no growth or stricture. Computed tomography ruled out any mass lesion or lymphadenopathy in that region.
|Figure 1: Barium swallow showing the fistulous communication between the lower end of the esophagus and the right lower lobe bronchus|
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|Figure 2: Esophagoscopy showing two fistulous openings in the lower esophagus|
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Patient was put on appropriate antibiotic therapy after obtaining culture sensitivity report. After a week of antibiotic therapy and chest physiotherapy, the sputum production reduced to 90 ml/day. On the day of surgery, he was premedicated with oral diazepam 5 mg and intramuscular glycopyrrolate 0.2 mg 45 min before surgery. On arrival to operating room, initially, Ryle's tube was inserted. Thereafter, under aseptic precautions and local analgesia, an epidural catheter was placed in the T 5−T 6 thoracic intervertebral space. Continuous monitoring of ECG, invasive blood pressure, end tidal carbon dioxide, urine output, and temperature was done. Rapid sequence induction was done using midazolam 2 mg, fentanyl 125 υg, and propofol 100 mg; succinylcholine 100 mg was administered to facilitate intubation with a 39 F (6.0 mm internal diameter) Robert Shaw left-sided double lumen tube (DLT) (Portex Ltd, UK). The position of the DLT was confirmed clinically, and by fiber-optic bronchoscope. The right lung was isolated and collapsed immediately to prevent gastric distention and to prevent loss of ventilation volume. The patient was then placed in the left lateral decubitus position and the placement of the DLT was reconfirmed. 900 ml of patient's blood was collected in bags containing CPDA solution and an equal volume of 6% Hetastarch solution was administered to maintain normovolemia. Patient's heart-rate and blood pressure were monitored closely throughout the collection. Anesthesia was maintained using nitrous oxide (N 2 O): oxygen (O 2 ) (50:50), isoflurane 0.5%, infusions of propofol 3 mg/kg/h, vecuronium 3 mg/h, and epidural infusion of 0.0625% bupivacaine with fentanyl 2 μg/ ml infused @8 ml/ h. Intra-operatively, the right lung was kept collapsed to aid dissection. Arterial blood gas analysis was done at every 30-minute interval and inspiratory O 2 and ventilator parameters were adjusted to keep SpO 2 between 90 and 100 mmHg and PaCO 2 between 45 and 50 mmHg.
Right posterolateral thoracotomy was done through the bed of the fifth rib. Gentle dissection near the inferior pulmonary ligament revealed a 1 cm × 2 cm fistulous tract communicating the right lower bronchus with lower esophagus and cystic changes in the right lower lobe. The esophageal opening was closed in two layers using interrupted absorbable sutures, and the right lower lobectomy was completed. Chest was closed after hemostasis with under-water seal drainage. At the end of the surgery, DLT was replaced with an 8 mm endotracheal tube. The patient was transferred to intensive care unit and was given ventilatory support. After 3 hours, when his electrolytes and temperature were normal, residual muscle relaxation was reversed and he was extubated. Epidural infusion was continued for the next 48 hours. Chest physiotherapy was continued postoperatively. He was discharged on the seventh postoperative day and regular follow-up showed significant improvement in his symptoms and the quality of life.
| Discussion|| |
Benign BEF can be congenital or acquired. Congenital BEF was first reported by Negus in 1929.  The congenital BEF is most commonly diagnosed in infancy. Very rarely they progress into adulthood when there is no associated oesophageal atresia; only 200 cases have been reported in the literature. , Diagnosis in these cases can be difficult because of the nonspecific nature of the symptoms. Benign BEF can remain undiagnosed for years, as is the case with our patient. Bouts of coughing while swallowing liquids (Ohno's sign) is pathognomonic of this condition and is present in 65% of the cases. , Conventional barium esophagography is considered the gold standard for diagnosing BEF. , CT scan may be utilized to rule out the presence of a neoplasm, adenopathy suggestive of TB and to define the extent of a coexisting pulmonary disease which may need resection. ,, Braimbridge and Keith  classified congenital BEF into four types. Type II (simple broncho-esophageal fistula) is the most prevalent variety and comprises almost 90% of all cases in some series.  Our patient had a type III fistula (Bronchoesophageal fistula with an intralobar cyst). The insidious nature of such a fistula may become life threatening with repeated infections leading to pneumonia, bronchiectasis, and abscess formation. , Despite the benign nature of this anomaly, if left untreated, it may lead to fatal complications.
The perioperative management aims to minimize the respiratory tract infection and to prevent the contamination of the fistulous tract. This is achieved by appropriate antibiotic therapy and adequate chest physiotherapy. This method significantly reduces sputum production as seen in our case. Intraoperative management aims to prevent gastric distension, maintain oxygenation throughout the surgery, and prevent known complications like surgical field fire. Because of low-resistance communication between bronchus and esophagus, leakage of gas into the stomach causes distension and jeopardizes ventilation. This can be avoided by prior placement of a nasogastric tube  and lung separation immediately after intubation. The isolation of the lung also prevents normal lung contamination and surgical field fire. Surgical field fire can further be avoided by collapsing the pathological lung and avoiding the oxygen insufflation to the pathological lung. , During one lung ventilation, oxygenation is ensured by providing positive end expiratory pressure to the dependent lung and ventilation is maintained by providing tidal volume of 8 to 10 ml/kg, and by adjusting the respiratory rate to keep arterial carbon dioxide less than 50 mmHg.
Postoperatively, it is essential to prevent collapse of the operated lung. This can be achieved by early extubation, incentive spirometry, and chest physiotherapy. Adequate analgesia in the postoperative period is helpful for patient co-operation. In our patient, we provided postoperative analgesia with the help of thoracic epidural with bupivacaine.
To conclude, it is essential to have a high index of suspicion to diagnose congenital BEF in adults. Anesthesiologists play an important role in successful management of these cases. If treated early, patient's quality of life can be improved to a great extent.
| 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. |
|2.||Cossentino MJ, Ormseth EJ, Tavaf-Motamen H, Cheney CP. Congenital bronchoesophageal fistula in the adult. A case report. Am J Gastroenterol 2000;95:2116-8. |
|3.||Linnane BM, Canny G. Congenital broncho-esophageal fistula: A case report. Respir Med 2006;100:1855-7. |
|4.||Kim JH, Park KH, Sung SW, Rho JR. Congenital bronchoesophageal fistulas in adult patients. Ann Thorac Surg 1995;60:151-5. |
|5.||Chiu HH, Chen CM, Mo LR, Chao TJ. Gastrointestinal: Tuberculous bronchoesophageal fistula. J Gastroenterol Hepatol 2006;21:1074. |
|6.||Ford MA, Mueller PS, Morgenthaler TI. Bronchoesophageal fistula due to broncholithiasis: A case. Respir Med 2005;99:830-5. |
|7.||Braimbridge MV, Keith HI. Oesophago-bronchial fistula in the adult. Thorax 1965;20:226-33. |
|8.||Rämö OJ, Salo JA, Mattila SP. Congenital bronchoesophageal fistula in the adult. Ann Thorac Surg 1995;59:887-9. |
|9.||Aguiló R, Minguella J, Jimeno J, Puig S, Galeras JA, Gayete A, et al. Congenital bronchoesophageal fistula in an adult woman. J Thorac Cardiovasc Surg 2006;131:916-7. |
|10.||Serrano N, Cortes JL. An unusual presentation of tracheoesophageal fistula. Intensive Care Med 1996;22:717-8. |
|11.||Singla AK, Campagna JA, Wright CD, Sandberg WS. Surgical field fire during a repair of bronchoesophageal fistula. Anesth Analg 2005;100:1062-4. |
|12.||Bruley ME. Surgical fires: Perioperative communication is essential to prevent this rare but devastating complication. Qual Saf Health Care 2004;13:467-71. |
R V Ranjan
House no 11, Type D II quarters, JIPMER campus, Puducherry-605 006
Source of Support: None, Conflict of Interest: None
[Figure 1], [Figure 2]