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CASE REPORT Table of Contents   
Year : 2011  |  Volume : 8  |  Issue : 2  |  Page : 244-248
Multiple anastomotic complications following repair of oesophageal atresia with tracheoesophageal fistula: A report of two cases


Department of Paediatric Surgery, Lady Hardinge Medical College and Kalawati Saran Children's Hospital, New Delhi - 110 001, India

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Date of Web Publication14-Oct-2011
 

   Abstract 

This report describes the clinical course and management of two children with multiple anastomotic complications following primary repair of oesophageal atresia (EA) with distal tracheoesophageal fistula (TEF). These included anastomotic leak and stricture, oesophageal perforation during antegrade dilatation, and finally a recurrent fistula between the oesophagus and the tracheo-bronchial tree. Ultimately, after multiple operative interventions, a successful outcome was achieved in both patients with preservation of the native oesophagus. The anastomotic complications following primary repair of EA with TEF are discussed, with special reference to difficulties in the management of recurrent TEF.

Keywords: Anastomotic, oesophageal atresia, oesophageal dilatation, oesophageal perforation, recurrent, tracheoesophageal fistula

How to cite this article:
Jain P, Debnath PR, Jain V, Chadha R, Choudhury SR, Puri A. Multiple anastomotic complications following repair of oesophageal atresia with tracheoesophageal fistula: A report of two cases. Afr J Paediatr Surg 2011;8:244-8

How to cite this URL:
Jain P, Debnath PR, Jain V, Chadha R, Choudhury SR, Puri A. Multiple anastomotic complications following repair of oesophageal atresia with tracheoesophageal fistula: A report of two cases. Afr J Paediatr Surg [serial online] 2011 [cited 2019 Sep 18];8:244-8. Available from: http://www.afrjpaedsurg.org/text.asp?2011/8/2/244/86075

   Introduction Top


Major morbidity and mortality in operated cases of oesophageal atresia with/without tracheoesophageal fistula (EA/TEF) is often a consequence of the complications of oesophageal anastomosis, namely anastomotic stricture, anastomotic leak, and finally, recurrence of the communication. [1] This report describes the successful management of two children with multiple anastomotic complications following primary repair of EA with distal TEF. These included anastomotic leak and stricture, oesophageal perforation during antegrade dilatation, and finally a recurrent fistula between the oesophagus and the tracheo-bronchial tree. The cases presented here illustrate the difficulties and technical challenges in the management of multiple surgical complications following repair of EA with TEF.


   Case Reports Top


Case 1

A 2.3-kg, one-month-old boy, was brought with recurrent regurgitation after feeds. The child had undergone primary repair for EA with a distal TEF at birth in another institution. A postoperative oral contrast oesophagogram performed there had shown an anastomotic stricture with a localized leak. A repeat oesophagogram, on presentation at our institution, revealed a tight anastomotic stricture without any leak. Under general anaesthesia, antegrade oesophageal dilatation with graded gum-elastic bougies was attempted but was complicated by oesophageal perforation needing insertion of an intercostal tube drain on the left side. Subsequent development of a left-sided loculated empyema with mediastinitis necessitated drainage by a left-sided thoracotomy along with a Stamm's gastrostomy for feeding. The postoperative course was uneventful.

An oesophagogram performed four months later showed a short tight anastomotic stricture without any evidence of anastomotic leak or gastroesophageal reflux (GER). Through a right-sided thoracotomy via an intrapleural approach, the short (0.5 cm-long) oesophageal stricture was resected after a difficult mobilization of the oesophagus, and a wide, spatulated oesophago-oesophageal anastomosis performed. Unfortunately, an anastomotic leak became apparent by the sixth postoperative day which, however, healed with conservative treatment. At discharge, the child was able to take semisolid diet orally and was advised follow-up for regular oesophageal dilatations.

The patient, however, was not brought for follow-up for two and a half years but presented at the age of three years with complaints of choking after feeds, although he was able to accept semisolid feeds. Dye study revealed a pseudo-diverticulum close to the anastomotic site, communicating with the tracheo-bronchial tree [Figure 1]. A right-sided thoracotomy was again performed through the fourth intercostal space. Careful dissection of the oesophagus showed the diverticulum to be a localized collection within the lung parenchyma communicating with the right main bronchus about 1 cm from the carina [Figure 2]. The oesophagopulmonary communication was isolated and divided. The oesophageal end of this fistula was repaired with interrupted sutures of 5-0 Prolene (Ethicon). To isolate the suture line a pericardial flap was interposed. Antegrade oesophageal dilatation was possible till 24 Fr. size. The postoperative course was uneventful with no evidence of leak in the postoperative dye study. He required a single oesophageal dilatation six weeks after surgery. At five years' follow-up, the child is thriving and is largely asymptomatic but has occasional dysphagia to solids.
Figure 1: Case 1. Oral contrast oesophagogram showing the oesophagopulmonary-bronchial fistula

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Figure 2: Case 1. Diagrammatic representation of the operative findings

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Case 2

A 10-month-old boy was brought with complaints of increasing dysphagia for the past one month. The boy had been operated at three days age for EA with a distal TEF, and the postoperative period had been uneventful. A barium swallow showed a moderately severe anastomotic stricture and, under general anaesthesia, antegrade oesophageal dilatation was performed with gum-elastic bougies till size 14 Fr. Symptoms of dysphagia however persisted along with fever and mild respiratory distress. A repeat contrast oesophagogram showed localized pooling of contrast at the anastomotic site, suggesting that an unrecognized perforation of the oesophagus had occurred during dilatation. This was managed conservatively. However, although able to swallow liquids, the child continued to have dysphagia along with recurrent chest infections and a contrast study performed three months later revealed a short anastomotic stricture, marked dilatation of the upper oesophagus, and a diverticulum at the anastomotic site. By means of a right-sided thoracotomy, the stricture was resected and a wide, spatulated oesophago-oesophagostomy performed. The immediate postoperative period was uneventful. Postoperatively, dysphagia improved for about two months but started worsening thereafter. A Technetium sulphur-colloid GER scan revealed moderately severe GER and a Boix-Ochoa anti-GER surgery was performed along with oesophageal dilatation till size 20 Fr. [2] The child was subsequently lost to follow-up but readmitted two years later with severe dysphagia, the child being able only to swallow liquids, along with severe cough and choking after feeds. An oral contrast oesophagogram revealed an anastomotic stricture, and a recurrent TEF. The child was advised surgery but was again lost to follow-up. Two years later, with worsening of the same symptoms, the child was readmitted. Right upper lobe pneumonia was also present. After antibiotics and active chest physiotherapy for 10 days, a right-sided thoracotomy was performed. A tight anastomotic stricture was found along with a 4-mm-wide TEF located 5 mm above the stricture. After mobilization of the oesophagus above and below the stricture, the TEF was isolated, divided, and the two ends closed with interrupted 4-0 Vicryl (Ethicon). The anastomotic stricture was resected and a wide oesophago-oesophagostomy performed. The postoperative course was uneventful and, at discharge, the child was able to accept semisolids and soft solids easily. The necessity for prophylactic postoperative dilatations was emphasized to the parents but, again, the child was lost to follow-up for seven months and then brought, at the age of six years, with severe dysphagia and a tight short anastomotic stricture on a contrast study. Antegrade oesophageal dilatation with gum-elastic bougies was unsuccessful and resulted in a perforation of the oesophagus. This was managed conservatively and after recovery from the acute symptoms, the parents insisted on taking the child home. Three months later, when the child was brought with severe dysphagia and choking after feeds, a contrast oesophagogram showed an anastomotic stricture with a large recurrent TEF [Figure 3]. A nuclear GER-scan showed that there was no GER. A repeat thoracotomy was performed after subperiosteal resection of the fifth rib and, as earlier, the stricture was resected after mobilization of the oesophagus. The recurrent TEF, now at a fresh site, was divided with closure of the two ends with interrupted sutures of 4-0 Vicryl. Recovery was uneventful and the contrast oesophagogram showed a wide anastomosis [Figure 4]. Since then, the child is on regular two to four-weekly follow-up for the past one and a half years. Three-weekly antegrade oesophageal dilatations till size 34 Fr were performed for the first four postoperative months, starting from four weeks after surgery, but the child has not needed oesophageal dilatation for the past one year. He has a normal diet and has substantial weight gain. His only problem is early bronchiectasis of the right-upper lobe of the lung for which he is receiving medical management.
Figure 3: Case 2. Oesophagogram after second recurrence of the TEF. Arrowhead points to the short anastomotic stricture

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Figure 4: Case 2. Oesophagogram after final surgery

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   Discussion Top


Although survival in patients of EA with/without TEF has improved to more than 95% in specialized centres, anastomotic complications are frequent and remain important causes of postoperative morbidity. The incidence of anastomotic stricture following repair of EA/TEF appears to vary but recent reports suggest that a stricture requiring dilatation may be present in as many as 80% patients. [3],[4],[5],[6] Anastomotic leaks occur in up to 17% of patients and can have potentially significant long-term consequences. [4] While 95% resolve spontaneously or with pleural drainage, oesophageal stricture follows in 50% cases. [4],[7] In the first case reported here, both the primary oesophageal anastomosis as well as that performed after resection of the anastomotic stricture was complicated by a leak and subsequent development of a stricture. In the second patient, the very first complication was the development of an anastomotic stricture. As most of these strictures can be managed by oesophageal dilatations at regular intervals, patient compliance plays an important role. As seen in both our patients, poor compliance may alter the course of management.

Our report emphasizes the inherent risks of antegrade oesophageal dilatation with gum-elastic bougies. [4],[8] At our centre, this was necessitated by the absence of facilities for balloon dilatation of the oesophagus with/ without fluoroscopic control, a technique that has been shown to be safe and efficacious, as well as superior to traditional bougie dilatation. [8],[9],[10] In our second patient, antegrade dilatation was twice complicated by oesophageal perforation. A likely contributory factor is that repeated oesophageal surgeries would have rendered the anastomotic site more friable and weakened, thereby increasing the risk of perforation.

A recurrent TEF is one of the most challenging problems to diagnose and manage. The incidence of recurrent TEF has been reported to be about 10% in most series. [11],[12],[13] The true incidence may even be greater because of difficulty in establishing the diagnosis. [12] Various factors have been implicated in the formation of recurrent TEF. Recurrent TEF is more common after an anastomotic leak or, as in our patients, following oesophageal perforation during dilatation of an anastomotic stricture. [7],[14] A leak at the anastomotic site may result in formation of a local abscess which eventually erodes into both organs leading to formation of the fistula which subsequently develops an epithelial lining. [15] Our first case was unusual in that the local abscess following the anastomotic leak eroded the lung parenchyma leading to a lung abscess with subsequently an oesophago-pulmonary-bronchial communication. Two similar cases have been reported earlier in the literature. [14] Anastomotic dilatation is also one of the contributory factors in re-communication. As a result of repeated dilatations, mucosal tear, progressive weakening of the suture line, and periesophagitis may lead to recurrence of fistula. [16],[17]

The diagnosis of recurrent TEF is often very difficult. Besides a high index of suspicion on clinical grounds, a good contrast study or oesophagoscopy/bronchoscopy is needed for diagnosis. [11],1[8] In a recent report of 26 cases of recurrent TEF, the diagnosis was made by a contrast study in 24 patients. [19] In case of high suspicion it is advisable to perform repeated contrast studies. [11] A contrast oesophagogram successfully diagnosed the recurrent TEF in both our patients.

A recurrent TEF rarely closes spontaneously and typically requires surgical repair. [7] As in the case of our patients, a transpleural approach through the old fourth space incision on the right side is feasible and, in fact, our second patient underwent four thoracotomies through the same incision, a rib resection being necessary on only the last occasion. [11] The blood supply of the oesophagus is generally excellent and one can, if necessary, carry out extensive mobilization of the proximal and distal oesophagus. [20] Although not necessary in our patients, bronchoscopic placement of a catheter through the fistula may aid dissection. [19] Conventional management carries a risk of at least a 20% incidence of second recurrences. [21] In a study of 26 cases of recurrent TEF managed by open surgery, postoperative complications included seven anastomotic leaks, four strictures, and three recurrent fistulas. [19] A variety of techniques have been used to reduce the incidence of recurrent TEF including the interposition of pleura, azygos vein, and vascularized pericardial flaps between the trachea and oesophagus and avoiding damage to the distal oesophagus [4],[13],[19] Interposition of a pericardial flap has been suggested as the most appropriate surgical procedure, and was successfully used by us in one patient. [19],[21] In recent years, successful endoscopic management of recurrent TEF, using fibrin glue (Tissel with added aprotinin) with diathermy, or argon plasma coagulation has been reported. [22],[23],[24],[25] Although success rates of up to 85% have been reported, multiple attempts may be required. [22],[23] Others continue to report failure with these techniques. [26]

In conclusion, our cases illustrate the number and variety of problems that can result from an anastomotic stricture or an anastomotic leak and subsequent oesophageal stricture, following primary repair of EA with TEF. Patient compliance is essential to effectively manage postoperative complications. Antegrade dilatation of an anastomotic oesophageal stricture remains a high-risk procedure, especially when multiple oesophageal surgeries have been performed. Following resection of an anastomotic stricture, the chances of restricturing are high, even in the absence of GER, and prophylactic anastomotic dilatation is advisable. Repeated thoracotomies on the same side are feasible to gain access to the oesophagus which can withstand repeated mobilizations, probably because of its excellent blood supply. Ultimately, a successful outcome was achieved in our patients, showing that oesophageal preservation is possible even in the face of multiple postoperative complications.

 
   References Top

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15.Ashour M. Recurrence of tracheo-oesophageal fistula 32 years after primary repair. Thorax 1986;41:76-7.  Back to cited text no. 15
    
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21.Wheatley MJ, Coran AG. Pericardial flap interposition for the definitive management of recurrent tracheo-esophageal fistula. J Pediatr Surg 1992;27:1122-5.  Back to cited text no. 21
    
22.Gutiérrez San Román C, Barrios JE, Lluna J, Ibañez V, Hernández E, Ayuso L, et al. Long-term assessment of the treatment of recurrent tracheoesophageal fistula with fibrin glue associated with diathermy. J Pediatr Surg 2006;41:1870-3.  Back to cited text no. 22
    
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26.Broto J, Asensio M, Vernet JM, Giné C, Cedeño R. [Endoscopic treatment of tracheo-oesophageal fistulas: Fact or fiction?]. Cir Pediatr 2003;16:69-72.  Back to cited text no. 26
    

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Correspondence Address:
Vishesh Jain
D-9, Green Park Extension, New Delhi, India - 110 016
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Source of Support: None, Conflict of Interest: None


DOI: 10.4103/0189-6725.86075

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    Figures

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