Secondary Logo

Journal Logo

Original Articles: Gastroenterology

Anastomotic Strictures Following Esophageal Atresia Repair: A 20-year Experience With Endoscopic Balloon Dilatation

Antoniou, Dimitris; Soutis, Michael; Christopoulos-Geroulanos, George

Author Information
Journal of Pediatric Gastroenterology and Nutrition: October 2010 - Volume 51 - Issue 4 - p 464-467
doi: 10.1097/MPG.0b013e3181d682ac
  • Free

Abstract

Esophageal stricture is a serious complication in a variety of otherwise benign conditions in children, associated with a high grade of morbidity because of dysphagia, regurgitation, and failure to thrive. The most common cause of benign esophageal stenosis in children is an anastomotic stricture following repair of esophageal atresia (EA). It is estimated that 18% to 50% of the patients with EA repair develop an anastomotic stricture with subsequent risk of malnutrition and aspiration (1). Traditionally, dilatation was performed with bougies, but this procedure is associated with high complication rate and severe mucosal injury leading to scar and additional stricture formation. Although balloon dilatation, under fluoroscopic or endoscopic control, has been widely accepted because of its advantage over other techniques and the safety of the procedure is well documented, there are few reports dealing with the long-term results of using balloon catheter dilators in children with postoperative anastomotic strictures. Moreover, in children experience is limited and there is a considerable diversity of opinion regarding the optimal size of balloon, intervals between dilatations, and technique of dilatation.

The purpose of the present study was to evaluate the safety, effectiveness, and long-term clinical results of balloon dilatation in the treatment of anastomotic strictures following EA repair.

PATIENTS AND METHODS

We retrospectively reviewed the medical records of 59 pediatric patients with anastomotic esophageal stricture following EA repair who underwent endoscopic balloon dilatation at our institution during a 20-year period. In the present study we enrolled only children with primary definitive repair of EA and distal tracheoesophageal fistula (TEF); patients with other type of EA and/or a delayed esophageal reconstruction were excluded. All of the patients presented with dysphagia of varying degree, and the diagnosis was confirmed by flexible upper gastrointestinal endoscopy and/or esophagography, depending on the severity of symptoms. All of the procedures were carried out under general anesthesia with endotracheal intubation. The endoscopes used were the GF XP20 (Olympus Optical Co Ltd, Tokyo, Japan) and the EG 2470, EG 1870 (Pentax Optical Co Ltd, Tokyo, Japan).

Dilatation was performed initially using a single-diameter balloon catheter (Rigiflex OTW Microvasive, Boston Scientific Corporation, Natick, MA) and during the last 8 years an over-the-wire multidiameter balloon catheter (CRE; Microvasive, Boston Scientific). The endoscope was passed to the level of the stricture and the guidewire was passed through the biopsy channel of the endoscope. The location, contour, and diameter of the stricture were estimated and an adequate balloon size was selected (Fig. 1A). The endoscope was removed with the wire left in place, and the balloon catheter was positioned under fluoroscopic control with its midportion across the stricture. The size of the balloon depended on the age of the patient and the diameter of the esophageal lumen at the level of the stricture as confirmed fluoroscopically by the presence of a “waist” or “hourglass” deformity (Fig. 1B). The balloon was inflated with radiopaque contrast material to the recommended pressure for 60 seconds. Disappearance of the “waist” indicated a successful dilatation (Fig. 1C); persistence of part of the “waist” was an indication that the procedure was partially successful.

FIGURE 1
FIGURE 1:
Images from a 6-month-old girl with an anastomotic esophageal stricture secondary to EA repair. A, Endoscopic view of the stricture at the beginning of the procedure. B, An 8–10 mm multidiameter balloon was placed. Anteroposterior radiograph shows the “waist” representing the stricture. C, The “waist” disappeared after successful balloon dilatation, indicating prompt termination of the procedure. D, Check endoscopy after successful balloon dilatation demonstrating minor mucosal injury. E, A 9-month follow-up endoscopy shows no stricture. The patient is symptom-free on a 13-month follow-up.

When the operator determined that an additional dilatation with a larger balloon was necessary, the inflation was repeated using a larger balloon catheter. After balloon retraction a check endoscopy was performed (Fig. 1D), and the patient fasted for 3 to 4 hours. Usually the procedure lasted 15 to 20 minutes. Antibiotics were not given routinely. When no complication occurred and oral intake was ascertained, the patient was discharged usually on the same day. The dilatation was repeated at intervals of 15 days on a day-clinic basis as long as the stricture kept recurring and then less frequently until the diameter remained steady (Fig. 1E). Thereafter, endoscopic dilatation was scheduled only if symptoms of dysphagia recurred.

Data obtained from patient's chart included sex, age, dysphagia grade, presence of gastroesophageal reflux (GER), number of dilatations, location of stricture, and complications. Dysphagia was graded on a 4-point scale: 0, no dysphagia; 1, unable to swallow certain solids or food impaction; 2, able to swallow soft foods; 3, only liquids are tolerated; 4, unable to swallow liquids. For infants younger than 8 months, dysphagia grade was based on endoscopic or esophagographic findings. The treatment was considered effective when dysphagia was grade 0 or 1 for >12 months after the last dilatation session. Depending on the effectiveness, the children were divided into 2 groups: group A, in which treatment was effective, and group B, in which treatment was ineffective. Response to dilatation was considered excellent when there was no need for any additional dilatation for recurrent strictures, satisfactory when up to 5 dilatations were required, and fair when >5 sessions were performed. If a patient had symptoms suggesting GER or when there were endoscopic signs of GER, then an 18-hour pH monitoring was carried out to assess the severity of GER, and an antireflux medical treatment was started.

Statistical Analysis

Data are expressed as mean ± 1 standard deviation for continuous variables and as incidence and percentages for categorical data. The Kolmogorov-Smirnov test was used for normality analysis of the parameters. Comparisons of continuous variables were analyzed using the Student t test and the Mann-Whitney nonparametric test, as appropriate. Comparisons of categorical variables were performed using the Pearson χ2 test and the Fisher exact test, as necessary. The Pearson correlation coefficient and the Spearman rho were used to assess bivariate linear correlations between variables. Differences were considered statistically significant if the null hypothesis could be rejected with >95% confidence (P < 0.05).

RESULTS

Between January 1988 and January 2008, 196 neonates with EA and/or TEF were treated in our hospital, with distal TEF being the most common type present in 167 cases (Table 1). During this period a total of 59 patients were treated with balloon dilatation for anastomotic stricture following EA (type III) repair. Forty-eight (28.7%) patients were treated for EA in our hospital, whereas 11 patients had primary repair in other institutions and were referred to us for management of the anastomotic stricture. There were 33 boys and 26 girls with a mean age of 10.5 months (range 1–36 months). Balloon dilatation was successful in all of the patients after a total of 165 sessions, ranging from 1 to 9 dilatation sessions (mean 2.79 ± 2.1) per patient. Eleven patients presented with grade 1 dysphagia (18.6%), 33 with grade 2 (55.9%), and 15 with grade 3 (25.4%). Response to dilatation was excellent in 21 cases (35.6%), satisfactory in 26 (44.1%), and fair in 12 (20.3%). There was a strong correlation between the number of sessions and the age (r = 0.71, P < 0.001) and between the grade of dysphagia at presentation and the number of sessions required for effective treatment (r = 0.8, P < 0.001). The treatment was effective in 47 patients (79.7%) and ineffective in 12 (20.3%). The mean number of sessions required for achieving relief of dysphagia in group A was 1.8 ± 0.8 (range 1–3), and the mean dysphagia grade was 1.9 ± 0.6. In the ineffective group B, the number of sessions was 6.6 ± 0.9 (range 6–9), and the dysphagia grade was 2.9 ± 0.3. There was a significant difference in the rate of effective treatment between patients with dysphagia grade <2 (44/45, 97.7%) and those with grade >2 (4/15, 26.6%, P < 0.001). All of the patients who achieved adequate dilatation were younger (mean 8.6 ± 6.8 months) in comparison to those unresponsive or partially responsive to balloon dilatation (mean 17.9 ± 4.7 months). GER was noted in 23 patients (39%), all of them treated initially by antireflux medication. The stricture was located in the middle third of the esophagus in 54 patients (91.5%) and in the upper third in 5 patients (8.5%). During the mean follow-up period of 19.5 months (range 12–36 months) after the last balloon dilatation session, none of the 47 children showed recurrent symptoms, and all are doing well. The treatment was not effective in 12 patients; all of them presented an associated GER. Four of these underwent surgery because of severe GER unresponsive to antireflux medical treatment. In 1 child a retrievable stent was placed, and 4 months later, the patient showed dysphagia only to some solid foods (grade 1). No major complication such as esophageal perforation or massive hemorrhage occurred during the procedures. All of the patients recovered well and were discharged on the same day. There were no procedure-related mortalities.

TABLE 1
TABLE 1:
Characteristics of patients with regard to type of atresia, sex, operative procedure, postoperative anastomotic leak, and stricture

DISCUSSION

Since the initial reported use of balloon catheters to treat esophageal strictures in children in 1984 (2,3), balloon dilatation has been used to treat strictures caused by reflux esophagitis, EA repair, caustic ingestion, sclerotherapy, achalasia, restrictive Nissen fundoplication, and interposition surgery (1–7).

Anastomotic stricture following EA repair is the most common cause of esophageal stenosis in children and infants, occurring in 18% to 50% of patients (1,5). In our series strictures following EA (type III) repair occurred in 28.7% of cases. The factors implicated in the pathogenesis of esophageal strictures include tension anastomosis, a 2-layer anastomosis, anastomotic leak, and GER. Endoscopic balloon dilatation using a Gruntzig or CRE balloon catheter over a guide wire is related to a lower complication rate because balloon catheters exert a direct radial dilating force equally and simultaneously on the stricture compared with the higher shearing forces of traditional bougienage technique (1,8,9). Moreover, CRE balloon catheter allows 3 progressively larger diameters of a single balloon and, at its maximum diameter, delivers significantly greater dilating force compared with the conventional balloon dilators (10). Our English-language literature review could identify only 14 studies with a cumulative total of 288 patients that report the use of balloon catheter dilatation in the treatment of esophageal stricture secondary to EA repair (Table 2). Few reports deal with the long-term efficacy of balloon dilatation in children with EA repair. Most authors consider esophageal dilatation to be successful when dysphagia declines and symptoms disappear, although recurrence is a common deficiency of this treatment modality. About half of the patients presented with esophageal stricture after EA repair will improve within 6 months by dilatation and 30% will show persistent stricture requiring repeated dilatations (20). One study (19) reported symptom relief in 93% of the patients 1 year after the last dilatation, whereas Lisy et al (17) confirmed an 80% efficacy. In another study, Said et al (13) reported a 100% procedural success rate for endoscopic balloon dilatation of anastomotic strictures after repair of EA in children. These results are similar to those of our study (79.7%), supporting that balloon dilatation is effective and safe first-line therapy for the management of anastomotic strictures following EA repair. There was a correlation between dysphagia grade at clinical presentation and the number of sessions required to achieve adequate dilatation. Patients who were able to swallow liquids (grade 3) before treatment were dilated for a mean of 5.5 sessions in comparison to 1.8 sessions among patients with mild dysphagia (grades 2 and 1). One year after the last dilatation 97.7% of children presented with dysphagia grade 1 or 2 were asymptomatic in contrast to 26.6% of those with higher-grade dysphagia. Ko et al (19) reported their experience on 29 children treated with balloon dilatation for anastomotic esophageal stricture secondary to surgical repair of EA. In their study 59% of the patients achieved relief from symptoms after a single balloon dilatation, compared with 45.4% reported by Lang et al (1) and the 35.6% of our study.

TABLE 2
TABLE 2:
Literature review on balloon dilatation in children with esophageal stricture following EA repair

GER is common in infants after surgical treatment of EA; its incidence varies widely from 18% to 70% and is thought to be a significant contributing factor in the formation of resistant strictures and even in the genesis of postoperative stricture (13,21,22). In the present study the incidence of GER was 39%, similar to that reported by Said et al (13), and the number of balloon dilatation sessions was significantly lower in patients without GER (1.4–4.8 per patient, respectively). Abnormalities of vagal nerve innervation, shortening of the intraabdominal esophagus due to extensive mobilization of the lower esophagus, anastomosis under tension, and abnormal peristaltic activity of the esophagus are factors related to GER in this group of infants (22). GER, whenever present, should be treated to preserve the results of the dilatation treatment and to minimize the number of dilatation sessions (21). A resistant stricture may be because of severe GER, and early recognition and control of this condition is essential in the management of these strictures. Patients unresponsive to antireflux medical treatment may require surgery or a temporary plastic self-expandable stent placement, although the usefulness of these stents in children is still unclear. It is well documented that laparoscopic antireflux surgery is an appropriate way to treat GER refractory to medical treatment in patients operated on for EA at birth (23). After surgical treatment, the majority of strictures require few dilatation procedures to resolve (21). It is believed that in patients who were older, the dense scarring due to long duration of symptoms may be the reason for a lower rate of success. Early detection of esophageal stricture and immediate initiation of balloon dilatation may prevent scar formation (1). Koivusalo et al (24,25) showed that the wait-and-see policy based on clinical indications is superior to routine dilatations, and >50% of the patients do not require dilatations at all. In addition, dilatation <3 weeks postoperatively may be too early in the healing process and could put the anastomosis at risk for perforation (12). In the present study the time of first dilatation varied, but in all of the cases therapy started as early as 4 weeks postoperatively. Patients younger than 6 months in this series required fewer dilatation sessions; the 70.9% of our study patients younger than 6 months of age required only 1 dilatation to achieve relief from dysphagia, and the 29.1% required 2 dilatations without any additional dilatation session needed to maintain symptomatic relief. These results are similar to those reported by Ko et al (19), in which relief was achieved in 70.6% of patients of similar age after 1 dilatation, whereas 11.6% of cases required >2 sessions. Esophageal perforation is the most serious, life-threatening complication of esophageal stricture dilatation. The rate of esophageal perforation related to balloon dilatation is reported to be 0% to 10% (4,6,11,19). In our series there was no perforation in 165 dilatation procedures; only blood staining on the balloon catheter occurred commonly after dilatation, indicating that mucosal disruption must be rather insignificant. The principal limitation of our study is because of its retrospective fashion. Even if the patient is correctly studied, data regarding dysphagia may not be precise because referring physicians and parents are involved in deciding when symptoms of dysphagia recur. In neonates and infants dysphagia grade was based on clinical symptoms and endoscopic findings because in this age group patients were fed exclusively with liquid meals. A standard study protocol would benefit from controlling the age of the patient, the severity of GER, and the degree of dysphagia.

Based on the results of the present study, esophageal balloon dilatation is recommended as the best primary treatment for anastomotic strictures following repair of EA because it is associated with a low complication rate and a good clinical outcome. In patients with stricture resistant to balloon dilatation, temporary placement of a covered retrievable stent can be considered. Laparoscopy is recommended for patients unresponsive to antireflux medical treatment and stent placement. The results of this report are consistent with previous studies supporting that endoscopic balloon dilatation is an effective, safe, and minimally invasive technique.

Acknowledgment

The authors thank Niki Spirtou for assistance with statistical analysis.

REFERENCES

1. Lang T, Hummer HP, Behrens R. Balloon dilatation is preferable to bougienage in children with esophageal atresia. Endoscopy 2001; 33:329–335.
2. Ball WS, Strife JL, Rosenkrantz J, et al. Esophageal strictures in children: treatment by balloon dilatation. Radiology 1984; 150:263–264.
3. Goldthorn JF, Ball WS Jr, Wilkinson LG, et al. Esophageal strictures in children: treatment by serial balloon catheter dilatation. Radiology 1984; 153:655–658.
4. Tam PKH, Sprigg A, Cudmore RE, et al. Endoscopy-guided balloon dilatation of esophageal strictures and anastomotic strictures after esophageal replacement in children. J Pediatr Surg 1991; 26:1101–1103.
5. Allmendinger N, Hallisey MJ, Markowitz SK, et al. Balloon dilatation of esophageal strictures in children. J Pediatr Surg 1996; 31:334–336.
6. Sandgren K, Malmfors G. Balloon dilatation of esophageal strictures in children. Eur J Pediatr Surg 1998; 8:9–11.
7. Lan LCL, Wong KKY, Lin SCL, et al. Endoscopic balloon dilatation of esophageal strictures in infants and children: 17 years' experience and a literature review. J Pediatr Surg 2003; 38:1712–1715.
8. Chetcuti P, Phelan PD. Gastrointestinal morbidity and growth after repair of oesophageal atresia and tracheo-esophageal fistula. Arch Dis Child 1993; 68:163–166.
9. Mclean GK, Leveen RF. Sheer stress in the performance of oesophageal dilatation: Comparison of balloon dilatation with bougienage. Radiology 1989; 172:983–986.
10. Goldstein JA, Barkin JS. Comparison of the diameter consistency and dilating force of the controlled radial expansion balloon catheter to the conventional balloon dilators. Am J Gastroenterol 2000; 95:3423–3427.
11. Fasulakis S, Andronikou S. Balloon dilatation in children for oesophageal strictures other than those due to primary repair of oesophageal atresia, interposition or restrictive fundoplication. Pediatr Radiol 2003; 33:682–687.
12. Kim IO, Yeon KM, Kim WS, et al. Perforation complicating balloon dilation of esophageal strictures in infants and children. Radiology 1993; 189:741–744.
13. Said M, Mekki M, Golli M, et al. Balloon dilatation of anastomotic strictures secondary to surgical repair of esophageal atresia. Br J Radiol 2003; 76:26–31.
14. Shah MD, Berman WF. Endoscopic balloon dilation of esophageal strictures in children. Gastrointest Endosc 1993; 39:153–156.
15. Yeming W, Somme S, Chenren S, et al. Balloon catheter dilatation in children with congenital and acquired esophageal anomalies. J Pediatr Surg 2002; 37:398–402.
16. Hoffer FA, Winter HS, Fellows KE, et al. The treatment of post-operative and peptic esophageal strictures after esophageal atresia repair. A program including dilatation with balloon catheters. Pediatr Radiol 1987; 17:454–458.
17. Lisy J, Hetkova M, Snajdauf J, et al. Long-term outcomes of balloon dilatation of esophageal strictures in children. Acad Radiol 1998; 5:832–835.
18. Weintraub JL, Eubig J. Balloon catheter dilatation of benign esophageal strictures in children. J Vasc Inter Radiol 2006; 17:831–835.
    19. Ko HK, Shin JH, Song HY, et al. Balloon dilatation of anastomotic strictures secondary to surgical repair of esophageal atresia in a pediatric population:long-term results. J Vasc Inter Radiol 2006; 17:1327–1333.
    20. Okada A, Usui N, Inoue M, et al. Esophageal atresia in Osaka: a review of 39 years' experience. J Pediatr Surg 1997; 32:1570–1574.
    21. Chittmittrapap S, Spitz L, Kiely EM, et al. Anastomotic stricture following repair of esophageal atresia. J Pediatr Surg 1990; 25:508–511.
    22. Tovar JA, Diez-Pardo JA, Murcia J, et al. Ambulatory 24-hour manometric and pH metric evidence of permanent impairment of clearance capacity in patients with esophageal atresia. J Pediatr Surg 1995; 30:1224–1231.
    23. Esposito C, Langer JC, Schaarschmidt K, et al. Laparoscopic antireflux procedures in the management of Gastroesophageal Reflux following esophageal atresia repair. J Pediatr Gastroenterol Nutr 2005; 40:349–351.
    24. Koivusalo A, Turunen P, Rintala RJ, et al. Is routine dilatation after repair of esophageal atresia with distal fistula better than dilatation when symptoms arise? Comparison of results of two European pediatric surgical centers. J Pediatr Surg 2004; 39:1643–1647.
    25. Koivusalo A, Pakarinen MP, Rintala RJ. Anastomotic dilatation after repair of esophageal atresia with distal fistula. Comparison of results after routine versus selective dilatation. Dis Esophagus 2009; 22:190–194.
    Keywords:

    balloon dilatation; esophageal atresia; stricture

    Copyright 2010 by ESPGHAN and NASPGHAN