|
 |
|
CASE REPORT |
|
|
|
| Year : 2023 | Volume
: 28
| Issue : 6 | Page : 523-525 |
| |
Successful management of a child with bronchoesophageal fistula associated with other anomalies
BV Raghunath1, Kavya Angadi2, AV Gurudutt3, Nidhi Srinivas4, L Manjunath5, SD Madhu6
1 Consultant Paediatric Surgeon, Rangadore Memorial Hospital, Bengaluru, Karnataka, India
2 Consultant Paediatric Anaesthetist, Rangadore Memorial Hospital, Bengaluru, Karnataka, India
3 Consultant and Chief Paediatric Intensivist, Rangadore Memorial Hospital, Bengaluru, Karnataka, India
4 Consultant Paediatric Intensivist, Rangadore Memorial Hospital, Bengaluru, Karnataka, India
5 Fellow in Paediatric Intensive Care, Rangadore Memorial Hospital, Bengaluru, Karnataka, India
6 Consultant Chief Radiologist, Rangadore Memorial Hospital, Bengaluru, Karnataka, India
| Date of Submission | 12-Apr-2023 |
| Date of Decision | 25-Jun-2023 |
| Date of Acceptance | 13-Jul-2023 |
| Date of Web Publication | 02-Nov-2023 |
Correspondence Address:
B V Raghunath
Rangadore Memorial Hospital, Bengaluru, Karnataka
India
 Source of Support: None, Conflict of Interest: None  | Check |
DOI: 10.4103/jiaps.jiaps_79_23
 Abstract | | |
Bronchoesophageal fistulas (BEFs) are rare malformations characterized by abnormal communication between the bronchus and the esophagus. Herein, we report a successful management of a child with BEF discovered as an intraoperative surprise and associated with the short gut, annular pancreas, and disorder of intestinal fixation.
Keywords: Annular pancreas, bronchoesophageal fistula, respiratory distress, short gut
How to cite this article:
Raghunath B V, Angadi K, Gurudutt A V, Srinivas N, Manjunath L, Madhu S D. Successful management of a child with bronchoesophageal fistula associated with other anomalies. J Indian Assoc Pediatr Surg 2023;28:523-5 |
How to cite this URL:
Raghunath B V, Angadi K, Gurudutt A V, Srinivas N, Manjunath L, Madhu S D. Successful management of a child with bronchoesophageal fistula associated with other anomalies. J Indian Assoc Pediatr Surg [serial online] 2023 [cited 2023 Nov 28];28:523-5. Available from: https://www.jiaps.com/text.asp?2023/28/6/523/389331 |
| Introduction | |  |
Bronchoesophageal fistulas (BEFs) are rare malformations characterized by abnormal communication between the bronchus and the esophagus. They are discrete from the regular tracheoesophageal fistulas encountered in neonates. They are usually detected when the child is being worked up for recurrent pneumonitis on contrast studies or computed tomography (CT) scans. Successful management of a child with BEF and associated with other anomalies has been described.
| Case Report | | |
A 3-year-old female child presented with a history of recurrent cough, hurried breathing, and abdominal distension for 4–5 h. A history of multiple episodes of nonbilious vomiting and progressive abdominal distension was noted for 5 h. The child had not passed stool for 1 day. There was no history of fever.
The child had a history of recurrent episodes of lower respiratory tract infection involving the right lung. Evaluation revealed right lower lobe congenital cystic adenomatoid malformation for which right lower lobectomy was done 2 years ago. The child had postoperative right chylothorax, which was treated conservatively.
The child remained asymptomatic for 2–3 months. Later, the child had intermittent episodes of nonbilious vomiting with mild abdominal distension once a month, which used to settle spontaneously. The child was evaluated by a medical gastroenterologist for the same with suspicion of malrotation with mid-gut volvulus. An upper gastrointestinal dye study was done which revealed duodenojejunal flexure on the left side with features of jejunal obstruction. Within 4–5 h of the study, the child had increasing abdominal distension, recurrent cough, and multiple episodes of nonbilious vomiting for which he was admitted.
At admission, the child was afebrile, tachycardic (180/min), and tachypneic (76/min) and had a SpO2 of 82% in room air. The child weighed 7.6 kg (<3rd centile). The child was drowsy but was responding to oral commands. Chest examination revealed intercostal/subcostal retractions with decreased air entry on the right side. Generalized abdominal distension was noted with visible step ladder peristalsis.
Initial blood gas showed moderate respiratory acidosis. Chest X-ray showed features of right lung pneumonitis. X-ray abdomen revealed a large dilated loop of small bowel involving the entire upper abdomen with a distal paucity of gas.
The child was electively intubated and stabilized. On intubation, bilious aspirates were noted in the endotracheal tube. Contrast-enhanced computerized tomography (CECT) scan of the chest and abdomen [Figure 1] revealed right upper and middle lobar pneumonia with malrotation and twisting at the root of the mesentery with dilated proximal bowel loops.
Laparotomy was done using a supraumbilical transverse incision after adequate stabilization. Features of disorders of cecal segment fixation were noted with duodenojejunal flexure on the left side of the spine with multiple inter-bowel adhesions. Cecum was free and was lying on the left side. No classical Ladd's bands were noted. The small bowel length was 100 cm. Closed loop obstruction of a segment of proximal dilated jejunum was noted. The annular pancreas was incidentally discovered.
Release of the adhesive bands with resection anastomosis of the dilated loop of jejunum with duodenoduodenostomy and appendectomy was done. However, during the duodenoduodenostomy procedure, an intermittent gush of air was noted with each breath given from the ventilator. Hence, a telephonic consultation was done with the radiologist to review the CT scan to see any abnormal communication with the respiratory tract. A definite fistulous communication between the right bronchus and the gastroesophageal junction was noted. Following this, the incision was extended vertically in the midline. A large, thick cartilaginous fistulous tract was noted extending from the right hemithorax up to the gastroesophageal junction [Figure 2].
Fistula was divided and the ends were sutured with 3-0 prolene. The leakage of air subsided and the abdomen was closed. The postoperative course was uneventful, and the child was extubated on postoperative day 2 and was on full feeds by postoperative day 4. The child was discharged on postoperative day 6.
The child is on follow-up for the last 6 months and is doing well. Cough and abdominal distension have subsided. The child is passing normal consistency stools 2–3 times/day and gaining weight.
| Discussion | | |
BEFs are rare disorders characterized by abnormal fistulous communication between the respiratory tract (bronchus or lung lobe) and the esophagus. They are reported more frequently in adults and may be due to benign or malignant causes.
In children, they are usually congenital but may be acquired.[1] They are usually characterized by chronic cough, recurrent respiratory tract infections, and failure to thrive.
They are usually diagnosed by an upper gastrointestinal contrast study. Rarely, a CECT chest or bronchoscopy may be needed.[2] In our case, it turned out to be an intraoperative surprise.
In 1965, Braimbridge and Keith[3] and Keith classified these anomalies into four subtypes based on the anatomy of the fistulous tract. In the first three types, the respiratory tract was normal, and in type 4, the fistula was connected to a sequestered lung segment. In our case, we could not identify the type as the child had already undergone a right lower lobectomy.
Smith[4] explained the embryologic basis of these anomalies and opined that they result from an incoordinated separation between the tracheal bud and the esophagus. Fistulous communication between the respiratory tract and the upper or middle one-third of the esophagus is reported.[2],[5] However, in our case, the attachment was at the gastroesophageal junction. The absence of any inflammatory features and thick cartilaginous fistulous tract mostly pointed toward a congenital cause in our case.[2]
The association of a short gut (small bowel measured only 100 cm), annular pancreas, and disorder of bowel fixation was peculiar in our case and has not been reported previously.
| Conclusion | | |
BEFs are rare lesions and need to be considered one of the differential diagnoses in children presenting with recurrent respiratory tract infections.
Declaration of patient consent
The authors certify that they have obtained all appropriate patient consent forms. In the form, the patient(s) has/have given his/her/their consent for his/her/their images and other clinical information to be reported in the journal. The patients understand that their names and initials will not be published and due efforts will be made to conceal their identity, but anonymity cannot be guaranteed.
Financial support and sponsorship
Nil.
Conflicts of interest
There are no conflicts of interest.
| References | | |
| 1. | Wang ZM, Zhang SC, Teng X. Esophageal diverticulum serves as a unique cause of bronchoesophageal fistula in children: A case report. Medicine (Baltimore) 2017;96:e9492.  |
| 2. | Risher WH, Arensman RM, Ochsner JL. Congenital bronchoesophageal fistula. Ann Thorac Surg 1990;49:500-5. |
| 3. | Braimbridge MV, Keith HI. Oesophago-bronchial fistula in the adult. Thorax 1965;20:226-33. |
| 4. | Smith DC. A congenital broncho-oesophageal fistula presenting in adult life without pulmonary infection. Br J Surg 1970;57:398-400. |
| 5. | Martins A, Gonçalves Á, Almeida T, Fazeres F, Midões A. Bronchoesophageal fistula in the setting of tuberculosis infection. J Gastrointest Surg 2017;21:2119-21. |
[Figure 1], [Figure 2]
|
