ORIGINAL ARTICLE: GASTROENTEROLOGY
Endoscopic Incision for the Treatment of Refractory Esophageal Anastomotic Strictures in Children Yuyong Tan, Jie Zhang, Junfeng Zhou, Tianying Duan, and Deliang Liu ABSTRACT Objectives: The aim of the present study was to assess the safety and efficacy of endoscopic incision (EI) for the treatment of refractory anastomotic esophageal strictures in pediatric patients. Methods: We retrospectively reviewed the medical records of pediatric patients with refractory anastomotic strictures after surgical repair of esophageal atresia who underwent 3 sessions of endoscopic treatments (dilation and/or stenting). They were treated with EI alone or together with esophageal stenting. Efficacy and safety were evaluated during periodical follow-up. Results: All of the 7 children received the procedure successfully with the operation time of 15 to 60 minutes. Four of them received EI alone, whereas the other 3 received EI with esophageal stenting (EIES). The symptoms remitted in all of the patients, and the dysphagia score decreased from 3–4 to 0–1 during follow-up from 1 to 21 months. The average diameter of stricture was enlarged from 3 mm (range 2–5 mm) to 10.6 mm (range 8–12 mm). One patient suffered from chest pain, which resolved within 3 days. Patient 1 had recurrence 11 months after EIES, and patient 6 had recurrence 3 months after EI. They all underwent an additional EI to maintain patency. No severe complications were observed during operation and periodical follow-up. Conclusions: EI is safe and appears effective for refractory esophageal anastomotic strictures in children in the short term. Large comparative studies are warranted to further confirm our findings. The long-term followup is necessary for assessing the long-term efficacy of the new technique. Key Words: anastomotic stricture, endoscopic treatment, esophageal atresia, esophageal stricture
(JPGN 2015;61: 319–322)
A
nastomotic stricture remains the most frequent complication after surgical repair of esophageal atresia (EA), with an incidence of 8% to 79% (1,2). Although most of them can be managed successfully with dilation, it is difficult to manage the refractory stricture that does not respond to repeated dilations (3,4). Affected patients often suffer from recurrent dysphagia and vomiting, which severely impair their quality of life and the ability to consume an adequate amount of food, and even result in growth retardation. Surgical resection is indicated in patients with established refractory stricture, but extensive injury and postoperative
Received January 24, 2015; accepted March 22, 2015. From the Department of Gastroenterology, Second Xiangya Hospital of Central South University, Changsha, Hunan, China. Address correspondence and reprint requests to Deliang Liu, Department of Gastroenterology, Second Xiangya Hospital of Central South University, Changsha 410011, China (e-mail: liudeliang@medmail. com.cn). The authors report no conflicts of interest. Copyright # 2015 by European Society for Pediatric Gastroenterology, Hepatology, and Nutrition and North American Society for Pediatric Gastroenterology, Hepatology, and Nutrition DOI: 10.1097/MPG.0000000000000801
JPGN
Volume 61, Number 3, September 2015
What Is Known
Most esophageal anastomotic strictures can be managed successfully with dilation, but some of them do not respond to repeated dilations. Surgical resection, intralesional steroid/mitomycin injection, and stenting are alternative methods for refractory stricture, but the efficacy remains unsatisfactory. Endoscopic incision is a novel treatment for refractory esophageal stricture with exciting results in adults.
What Is New
Our study has shown that endoscopic incision is safe and effective in the short term for refractory esophageal anastomotic stricture in children. Endoscopic incision plus esophageal stenting may help to maintain patency for strictures 1.5 cm.
restenosis often ensue (1,4). The efficacy of intralesional steroid/ mitomycin injection is still controversial (5–8). Although esophageal stent placement is an alternative method, the rate of migration is high, and the long-term efficacy remains unsatisfactory (9,10). Endoscopic incision (EI) is a novel technique for treating refractory esophageal stricture and has shown exciting results in adults (11– 13). Little is known about its efficacy, however, in pediatric patients. We herein report our preliminary results of treatment of pediatric refractory esophageal stricture with EI at our hospital.
METHODS Patient Information This retrospective study was approved by the ethics committee of the Second Xiangya Hospital of Central South University. The inclusion criteria for enrollment in the study were as follows: esophageal stricture after surgical repair of EA that was diagnosed on the basis of clinical symptoms, esophagogastroduodenoscopy, and barium meal; refractory stricture was considered because the stricture could not be improved to a diameter of 6 mm, or symptom of dysphagia was not relieved after 3 sessions of endoscopic treatment (ie, dilation or stenting) (1,3); the stricture was 1.5 cm, a fully covered retrievable metal stent with antireflux valve at the distal end was placed after EI. Figure 1 depicts an example of EI with esophageal stenting (EIES).
320
Postoperative Management Patients were kept nil per os for 2 days, on a liquid diet for 5 days, and returned gradually to a soft diet within a month. Intravenous proton pump inhibitor (PPI) and antibiotics were prophylactically used for 3 days. For patients who received EI, gastroscopy was scheduled in every 2 to 3 months within the first 6 months after the procedure. For patients who received EIES, a chest x-ray was done monthly to check for stent migration until stent removal, whereas endoscopy was performed to adjust, reset, or remove the stent. For all of the patients, endoscopy was performed once they have recurrent dysphagia. Stent removal was determined according to the following criteria: if there was no migration but the stent had been placed for as long as 12 weeks; because it fell into the stomach, but the dysphagia symptom was relieved; if there is obvious tissue overgrowth or ulceration on the upper and lower edges of the stent during surveillance endoscopy. The stent was reset when it shifted from the original position, but the stricture did not improve. Patients were asked to take oral PPI for 4 weeks.
Evaluation of Dysphagia Before and After EI We evaluated the grade of dysphagia symptom using the following dysphagia score (14): 0, able to eat a normal diet; 1, unable to swallow certain solids; 2, able to swallow semisolid foods; 3, able to swallow liquids only; 4, unable to swallow liquids. www.jpgn.org
Copyright 2015 by ESPGHAN and NASPGHAN. Unauthorized reproduction of this article is prohibited.
JPGN
Volume 61, Number 3, September 2015
Endoscopic Incision
TABLE 1. Clinical characteristics and treatment outcomes of the 7 patients
Patient no.
Sex/ age, y
Prior therapy
1
M/4
2
F/7
3
M/6
4
M/5
5
M/4
6 7
F/8 M/6
4 Dilations 2 Stenting 6 Dilations 3 Stenting 5 Dilations 1 Stenting 4 Dilations 4 Stenting 6 Dilations 4 Stenting 4 Dilations 5 Dilations 1 Stenting
Distance from the incisors, cm
Length of the stricture, cm
Diameter of stricture
Dysphagia score
Pre
Post
Pre
Post
Operation time, min
16 22 18
2.0 1.0 1.0
0.2 0.2 0.5
1.0 1.1 1.0
4
1
60
3
1
15
28
3.0
0.2
1.2
4
0
35
20
1.2
0.3
1.0
4
1
20
16
3.0
0.2
1.2
4
1
30
20 19
1.0 1.2
0.4 0.5
0.8 1.2
3 3
1 0
25 20
Stenting insertion
Complication/ recurrence
Follow-up, mo
Yes 13 105 No
Recurrent 11 mo later
21
None
18
Yes 14 85 No
None
12
Chest pain
9
Yes 13 105 No No
None
7
Recurrent 3 mo later None
6 1
Refers to the follow-up duration after stent removal.
Definition of Failure and Indication of Repeat Treatment Treatment failure was defined as the inability to pass an endoscope with a diameter of 5.9 mm and the dysphagia score >2. We performed additional EI/EIES treatment when the patients had treatment failure.
RESULTS Patient Characteristics Of the 7 patients, 5 were boys and 2 were girls, ages 4 to 8 years old. There were 8 strictures in the 7 patients. All of them had received several treatments before EI, the dysphagia score was 3 or 4. Detailed clinical data were presented in Table 1.
Treatment Outcome and Adverse Events EI/EIES was performed successfully in all of the patients. The average operation time was 32 minutes (range 15–60 minutes). Patient 1, 3, and 5 underwent EIES, and the stents were successfully removed 4 to 8 weeks after the procedure. Patients 2, 4, 6, and 7 underwent EI only. All of the patients were given fluid food 48 hours postoperatively. Sustained symptom improvement was achieved in 71.4% (5/7) of the patients during a follow-up of 1 to 21 months after a single treatment. The average diameter of stricture was enlarged from 3 (range 2–5 mm) to 10.6 mm (range 8–12 mm). The dysphagia score decreased to 0 or 1. Recurrences were noted 11 months after EIES in patient 1 and 3 months after EI in patient 6. They both underwent an additional EI to maintain patency. Patient 4 suffered from a mild chest pain that disappeared within 3 days without analgesic therapy (Table 1).
DISCUSSION In the present study, we described the use of EI for the treatment of refractory esophageal anastomotic stricture in children. To our knowledge, this is the first case series for the novel technique in pediatric patients. Endoscopic dilation remains the first-line therapy for esophageal strictures and has proved to be safe and effective. Refractory stricture refers to those who do not respond to repeated www.jpgn.org
dilations. Current strategies to manage refractory stricture mainly include surgical resection and endoscopic therapy. Surgical resection is indicated in patients with established refractory strictures but extensive injury and postoperative restenosis often ensue. Endoscopic methods such as local drug injection, stent, and incision are now introduced to clinical use. Intralesional steroid injection may reduce the risk of recurrent stricture formation because it can inhibit collagen formation, enhance collagen breakdown, and decrease fibrotic healing, which occur after dilation and prevent crosslinking of collagen that causes contraction in scar tissue. There have been only a few case reports, however, for intralesional steroid injection and EA patients (15–17), and no consensus on the optimal injection technique, frequency of injection, and dosage has been established. Although mitomycin may be helpful for refractory stricture by decreasing collagen synthesis and scar formation as shown in a few case reports (18–22), there is no benefit in the resolution of the stricture after surgical repair of EA when adding mitomycin-C treatment compared with repeated esophageal dilations alone in historical controls as shown by Chapuy et al (8). Esophageal stenting is an alternative method, but its complications, such as tissue overgrowth, migration, and pain, are not negligible (9,10). EI is a novel technique for the treatment of anastomotic stricture and has shown exciting results in adults (11–13). Little is known about its efficacy, however, in pediatric patients after surgical repair for EA. Tan et al (23) reported a patient successfully treated with EI and esophageal stenting. In the present case series, all of the patients had several previous attempts of dilation/stenting but failed to obtain a sustained symptomatic improvement. We successfully managed them with EI/EIES, with a sustained symptomatic improvement during a follow-up period of 1 to 21 months. Efficacy of EI was associated with the length of the stricture. Hordijk et al (11) found that all of the 12 patients with a stricture 1.5 cm. Stenting is superior to
321
Copyright 2015 by ESPGHAN and NASPGHAN. Unauthorized reproduction of this article is prohibited.
Liu et al
JPGN
dilation for the following 3 reasons. First, stenting offers tremendous potential advantages over dilation in its ability to provide continuous, radially oriented dilation pressures during a period of time, thus being conducive for scar remodeling and lowering the risks of perforation and restenosis. Second, fully covered stents can be used to protect the incision wound surface from gastric acid erosion, possibly reducing the incidence of recurrence. Third, endoscopic intervention should be performed under general anesthesia for pediatric patients (24). Dilation is recommended once a week after incision, for an average of 4 episodes. Stenting may be retained for >4 weeks, however, which reduces the frequency of invasive operation and the risk of operation-related complications. Limitations of the study include its small sample size, retrospective design, and relative short-term follow-up. In conclusion, the use of EI is feasible and safe for treating refractory esophageal stricture in pediatric patients and is effective in the short term. As these procedures may be associated with significant risks, they should be performed by an experienced expert who is capable of operating the endoscope skillfully and managing potential complications. Larger comparative studies are warranted to further confirm our findings, and long-term follow-up is necessary to assess the long-term efficacy of the new technique.
REFERENCES 1. Levesque D, Baird R, Laberge JM. Refractory strictures post-esophageal atresia repair: what are the alternatives. Dis Esophagus 2013;26: 382–7. 2. Baird R, Laberge JM, Levesque D. Anastomotic stricture after esophageal atresia repair: a critical review of recent literature. Eur J Pediatr Surg 2013;23:204–13. 3. Kochman ML, McClave SA, Boyce HW. The refractory and the recurrent esophageal stricture: a definition. Gastrointest Endosc 2005;62:474–5. 4. de Wijkerslooth LR, Vleggaar FP, Siersema PD. Endoscopic management of difficult or recurrent esophageal strictures. Am J Gastroenterol 2011;106:2080–91. 5. Berenson GA, Wyllie R, Caulfield M, et al. Intralesional steroids in the treatment of refractory esophageal strictures. J Pediatr Gastroenterol Nutr 1994;18:250–2. 6. Ramage JI Jr, Rumalla A, Baron TH, et al. A prospective, randomized, double-blind, placebo-controlled trial of endoscopic steroid injection therapy for recalcitrant esophageal peptic strictures. Am J Gastroenterol 2005;100:2419–25. 7. Berger M, Ure B, Lacher M. Mitomycin C in the therapy of recurrent esophageal strictures: hype or hope. Eur J Pediatr Surg 2012;22:109– 16.
322
Volume 61, Number 3, September 2015
8. Chapuy L, Pomerleau M, Faure C. Topical mitomycin-C application in recurrent esophageal strictures after surgical repair of esophageal atresia. J Pediatr Gastroenterol Nutr 2014;59:608–11. 9. Dan DT, Gannavarapu B, Lee JG, et al. Removable esophageal stents have poor efficacy for the treatment of refractory benign esophageal strictures (RBES). Dis Esophagus 2014;27:511–7. 10. Zhang J, Ren L, Huo J, et al. The use of retrievable fully covered selfexpanding metal stent in refractory postoperative restenosis of benign esophageal stricture in children. J Pediatr Surg 2013;48:2235–40. 11. Hordijk ML, Siersema PD, Tilanus HW, et al. Electrocautery therapy for refractory anastomotic strictures of the esophagus. Gastrointest Endosc 2006;63:157–63. 12. Muto M, Ezoe Y, Yano T, et al. Usefulness of endoscopic radial incision and cutting method for refractory esophagogastric anastomotic stricture (with video). Gastrointest Endosc 2012;75:965–72. 13. Simmons DT, Baron TH. Electroincision of refractory esophagogastric anastomotic strictures. Dis Esophagus 2006;19:410–4. 14. Atkinson M, Ferguson R, Ogilvie AL. Management of malignant dysphagia by intubation at endoscopy. J R Soc Med 1979;72:894–7. 15. Holder TM, Ashcraft KW, Leape L. The treatment of patients with esophageal strictures by local steroid injections. J Pediatr Surg 1969;4:646–53. 16. Zein NN, Greseth JM, Perrault J. Endoscopic intralesional steroid injections in the management of refractory esophageal strictures. Gastrointest Endosc 1995;41:596–8. 17. Gandhi RP, Cooper A, Barlow BA. Successful management of esophageal strictures without resection or replacement. J Pediatr Surg 1989;24:745–9. 18. Heran MK, Baird R, Blair GK, et al. Topical mitomycin-C for recalcitrant esophageal strictures: a novel endoscopic/fluoroscopic technique for safe endoluminal delivery. J Pediatr Surg 2008;43:815–8. 19. Uhlen S, Fayoux P, Vachin F, et al. Mitomycin C: an alternative conservative treatment for refractory esophageal stricture in children. Endoscopy 2006;38:404–7. 20. Rosseneu S, Afzal N, Yerushalmi B, et al. Topical application of mitomycin-C in oesophageal strictures. J Pediatr Gastroenterol Nutr 2007;44:336–41. 21. Daher P, Riachy E, Georges B, et al. Topical application of mitomycin C in the treatment of esophageal and tracheobronchial stricture: a report of 2 cases. J Pediatr Surg 2007;42:E9–11. 22. Chung J, Connolly B, Langer J, et al. Fluoroscopy-guided topical application of mitomycin-C in a case of refractory esophageal stricture. J Vasc Interv Radiol 2010;21:152–5. 23. Tan Y, Wang X, Liu D, et al. Endoscopic incision plus esophageal stenting for refractory esophageal stricture in children. Endoscopy (46 Suppl 1 UCTN):2014:E111–2. 24. Faccin G, Merlo F, Moretti T, et al. Anesthesiologic problems in transluminal balloon dilatation of esophageal stenosis in children. Minerva Anestesiol 1990;6:77–80.
www.jpgn.org
Copyright 2015 by ESPGHAN and NASPGHAN. Unauthorized reproduction of this article is prohibited.
Endoscopic Incision for the Treatment of Refractory Esophageal Anastomotic Strictures in Children Yuyong Tan, Jie Zhang, Junfeng Zhou, Tianying Duan, and Deliang Liu ABSTRACT Objectives: The aim of the present study was to assess the safety and efficacy of endoscopic incision (EI) for the treatment of refractory anastomotic esophageal strictures in pediatric patients. Methods: We retrospectively reviewed the medical records of pediatric patients with refractory anastomotic strictures after surgical repair of esophageal atresia who underwent 3 sessions of endoscopic treatments (dilation and/or stenting). They were treated with EI alone or together with esophageal stenting. Efficacy and safety were evaluated during periodical follow-up. Results: All of the 7 children received the procedure successfully with the operation time of 15 to 60 minutes. Four of them received EI alone, whereas the other 3 received EI with esophageal stenting (EIES). The symptoms remitted in all of the patients, and the dysphagia score decreased from 3–4 to 0–1 during follow-up from 1 to 21 months. The average diameter of stricture was enlarged from 3 mm (range 2–5 mm) to 10.6 mm (range 8–12 mm). One patient suffered from chest pain, which resolved within 3 days. Patient 1 had recurrence 11 months after EIES, and patient 6 had recurrence 3 months after EI. They all underwent an additional EI to maintain patency. No severe complications were observed during operation and periodical follow-up. Conclusions: EI is safe and appears effective for refractory esophageal anastomotic strictures in children in the short term. Large comparative studies are warranted to further confirm our findings. The long-term followup is necessary for assessing the long-term efficacy of the new technique. Key Words: anastomotic stricture, endoscopic treatment, esophageal atresia, esophageal stricture
(JPGN 2015;61: 319–322)
A
nastomotic stricture remains the most frequent complication after surgical repair of esophageal atresia (EA), with an incidence of 8% to 79% (1,2). Although most of them can be managed successfully with dilation, it is difficult to manage the refractory stricture that does not respond to repeated dilations (3,4). Affected patients often suffer from recurrent dysphagia and vomiting, which severely impair their quality of life and the ability to consume an adequate amount of food, and even result in growth retardation. Surgical resection is indicated in patients with established refractory stricture, but extensive injury and postoperative
Received January 24, 2015; accepted March 22, 2015. From the Department of Gastroenterology, Second Xiangya Hospital of Central South University, Changsha, Hunan, China. Address correspondence and reprint requests to Deliang Liu, Department of Gastroenterology, Second Xiangya Hospital of Central South University, Changsha 410011, China (e-mail: liudeliang@medmail. com.cn). The authors report no conflicts of interest. Copyright # 2015 by European Society for Pediatric Gastroenterology, Hepatology, and Nutrition and North American Society for Pediatric Gastroenterology, Hepatology, and Nutrition DOI: 10.1097/MPG.0000000000000801
JPGN
Volume 61, Number 3, September 2015
What Is Known
Most esophageal anastomotic strictures can be managed successfully with dilation, but some of them do not respond to repeated dilations. Surgical resection, intralesional steroid/mitomycin injection, and stenting are alternative methods for refractory stricture, but the efficacy remains unsatisfactory. Endoscopic incision is a novel treatment for refractory esophageal stricture with exciting results in adults.
What Is New
Our study has shown that endoscopic incision is safe and effective in the short term for refractory esophageal anastomotic stricture in children. Endoscopic incision plus esophageal stenting may help to maintain patency for strictures 1.5 cm.
restenosis often ensue (1,4). The efficacy of intralesional steroid/ mitomycin injection is still controversial (5–8). Although esophageal stent placement is an alternative method, the rate of migration is high, and the long-term efficacy remains unsatisfactory (9,10). Endoscopic incision (EI) is a novel technique for treating refractory esophageal stricture and has shown exciting results in adults (11– 13). Little is known about its efficacy, however, in pediatric patients. We herein report our preliminary results of treatment of pediatric refractory esophageal stricture with EI at our hospital.
METHODS Patient Information This retrospective study was approved by the ethics committee of the Second Xiangya Hospital of Central South University. The inclusion criteria for enrollment in the study were as follows: esophageal stricture after surgical repair of EA that was diagnosed on the basis of clinical symptoms, esophagogastroduodenoscopy, and barium meal; refractory stricture was considered because the stricture could not be improved to a diameter of 6 mm, or symptom of dysphagia was not relieved after 3 sessions of endoscopic treatment (ie, dilation or stenting) (1,3); the stricture was 1.5 cm, a fully covered retrievable metal stent with antireflux valve at the distal end was placed after EI. Figure 1 depicts an example of EI with esophageal stenting (EIES).
320
Postoperative Management Patients were kept nil per os for 2 days, on a liquid diet for 5 days, and returned gradually to a soft diet within a month. Intravenous proton pump inhibitor (PPI) and antibiotics were prophylactically used for 3 days. For patients who received EI, gastroscopy was scheduled in every 2 to 3 months within the first 6 months after the procedure. For patients who received EIES, a chest x-ray was done monthly to check for stent migration until stent removal, whereas endoscopy was performed to adjust, reset, or remove the stent. For all of the patients, endoscopy was performed once they have recurrent dysphagia. Stent removal was determined according to the following criteria: if there was no migration but the stent had been placed for as long as 12 weeks; because it fell into the stomach, but the dysphagia symptom was relieved; if there is obvious tissue overgrowth or ulceration on the upper and lower edges of the stent during surveillance endoscopy. The stent was reset when it shifted from the original position, but the stricture did not improve. Patients were asked to take oral PPI for 4 weeks.
Evaluation of Dysphagia Before and After EI We evaluated the grade of dysphagia symptom using the following dysphagia score (14): 0, able to eat a normal diet; 1, unable to swallow certain solids; 2, able to swallow semisolid foods; 3, able to swallow liquids only; 4, unable to swallow liquids. www.jpgn.org
Copyright 2015 by ESPGHAN and NASPGHAN. Unauthorized reproduction of this article is prohibited.
JPGN
Volume 61, Number 3, September 2015
Endoscopic Incision
TABLE 1. Clinical characteristics and treatment outcomes of the 7 patients
Patient no.
Sex/ age, y
Prior therapy
1
M/4
2
F/7
3
M/6
4
M/5
5
M/4
6 7
F/8 M/6
4 Dilations 2 Stenting 6 Dilations 3 Stenting 5 Dilations 1 Stenting 4 Dilations 4 Stenting 6 Dilations 4 Stenting 4 Dilations 5 Dilations 1 Stenting
Distance from the incisors, cm
Length of the stricture, cm
Diameter of stricture
Dysphagia score
Pre
Post
Pre
Post
Operation time, min
16 22 18
2.0 1.0 1.0
0.2 0.2 0.5
1.0 1.1 1.0
4
1
60
3
1
15
28
3.0
0.2
1.2
4
0
35
20
1.2
0.3
1.0
4
1
20
16
3.0
0.2
1.2
4
1
30
20 19
1.0 1.2
0.4 0.5
0.8 1.2
3 3
1 0
25 20
Stenting insertion
Complication/ recurrence
Follow-up, mo
Yes 13 105 No
Recurrent 11 mo later
21
None
18
Yes 14 85 No
None
12
Chest pain
9
Yes 13 105 No No
None
7
Recurrent 3 mo later None
6 1
Refers to the follow-up duration after stent removal.
Definition of Failure and Indication of Repeat Treatment Treatment failure was defined as the inability to pass an endoscope with a diameter of 5.9 mm and the dysphagia score >2. We performed additional EI/EIES treatment when the patients had treatment failure.
RESULTS Patient Characteristics Of the 7 patients, 5 were boys and 2 were girls, ages 4 to 8 years old. There were 8 strictures in the 7 patients. All of them had received several treatments before EI, the dysphagia score was 3 or 4. Detailed clinical data were presented in Table 1.
Treatment Outcome and Adverse Events EI/EIES was performed successfully in all of the patients. The average operation time was 32 minutes (range 15–60 minutes). Patient 1, 3, and 5 underwent EIES, and the stents were successfully removed 4 to 8 weeks after the procedure. Patients 2, 4, 6, and 7 underwent EI only. All of the patients were given fluid food 48 hours postoperatively. Sustained symptom improvement was achieved in 71.4% (5/7) of the patients during a follow-up of 1 to 21 months after a single treatment. The average diameter of stricture was enlarged from 3 (range 2–5 mm) to 10.6 mm (range 8–12 mm). The dysphagia score decreased to 0 or 1. Recurrences were noted 11 months after EIES in patient 1 and 3 months after EI in patient 6. They both underwent an additional EI to maintain patency. Patient 4 suffered from a mild chest pain that disappeared within 3 days without analgesic therapy (Table 1).
DISCUSSION In the present study, we described the use of EI for the treatment of refractory esophageal anastomotic stricture in children. To our knowledge, this is the first case series for the novel technique in pediatric patients. Endoscopic dilation remains the first-line therapy for esophageal strictures and has proved to be safe and effective. Refractory stricture refers to those who do not respond to repeated www.jpgn.org
dilations. Current strategies to manage refractory stricture mainly include surgical resection and endoscopic therapy. Surgical resection is indicated in patients with established refractory strictures but extensive injury and postoperative restenosis often ensue. Endoscopic methods such as local drug injection, stent, and incision are now introduced to clinical use. Intralesional steroid injection may reduce the risk of recurrent stricture formation because it can inhibit collagen formation, enhance collagen breakdown, and decrease fibrotic healing, which occur after dilation and prevent crosslinking of collagen that causes contraction in scar tissue. There have been only a few case reports, however, for intralesional steroid injection and EA patients (15–17), and no consensus on the optimal injection technique, frequency of injection, and dosage has been established. Although mitomycin may be helpful for refractory stricture by decreasing collagen synthesis and scar formation as shown in a few case reports (18–22), there is no benefit in the resolution of the stricture after surgical repair of EA when adding mitomycin-C treatment compared with repeated esophageal dilations alone in historical controls as shown by Chapuy et al (8). Esophageal stenting is an alternative method, but its complications, such as tissue overgrowth, migration, and pain, are not negligible (9,10). EI is a novel technique for the treatment of anastomotic stricture and has shown exciting results in adults (11–13). Little is known about its efficacy, however, in pediatric patients after surgical repair for EA. Tan et al (23) reported a patient successfully treated with EI and esophageal stenting. In the present case series, all of the patients had several previous attempts of dilation/stenting but failed to obtain a sustained symptomatic improvement. We successfully managed them with EI/EIES, with a sustained symptomatic improvement during a follow-up period of 1 to 21 months. Efficacy of EI was associated with the length of the stricture. Hordijk et al (11) found that all of the 12 patients with a stricture 1.5 cm. Stenting is superior to
321
Copyright 2015 by ESPGHAN and NASPGHAN. Unauthorized reproduction of this article is prohibited.
Liu et al
JPGN
dilation for the following 3 reasons. First, stenting offers tremendous potential advantages over dilation in its ability to provide continuous, radially oriented dilation pressures during a period of time, thus being conducive for scar remodeling and lowering the risks of perforation and restenosis. Second, fully covered stents can be used to protect the incision wound surface from gastric acid erosion, possibly reducing the incidence of recurrence. Third, endoscopic intervention should be performed under general anesthesia for pediatric patients (24). Dilation is recommended once a week after incision, for an average of 4 episodes. Stenting may be retained for >4 weeks, however, which reduces the frequency of invasive operation and the risk of operation-related complications. Limitations of the study include its small sample size, retrospective design, and relative short-term follow-up. In conclusion, the use of EI is feasible and safe for treating refractory esophageal stricture in pediatric patients and is effective in the short term. As these procedures may be associated with significant risks, they should be performed by an experienced expert who is capable of operating the endoscope skillfully and managing potential complications. Larger comparative studies are warranted to further confirm our findings, and long-term follow-up is necessary to assess the long-term efficacy of the new technique.
REFERENCES 1. Levesque D, Baird R, Laberge JM. Refractory strictures post-esophageal atresia repair: what are the alternatives. Dis Esophagus 2013;26: 382–7. 2. Baird R, Laberge JM, Levesque D. Anastomotic stricture after esophageal atresia repair: a critical review of recent literature. Eur J Pediatr Surg 2013;23:204–13. 3. Kochman ML, McClave SA, Boyce HW. The refractory and the recurrent esophageal stricture: a definition. Gastrointest Endosc 2005;62:474–5. 4. de Wijkerslooth LR, Vleggaar FP, Siersema PD. Endoscopic management of difficult or recurrent esophageal strictures. Am J Gastroenterol 2011;106:2080–91. 5. Berenson GA, Wyllie R, Caulfield M, et al. Intralesional steroids in the treatment of refractory esophageal strictures. J Pediatr Gastroenterol Nutr 1994;18:250–2. 6. Ramage JI Jr, Rumalla A, Baron TH, et al. A prospective, randomized, double-blind, placebo-controlled trial of endoscopic steroid injection therapy for recalcitrant esophageal peptic strictures. Am J Gastroenterol 2005;100:2419–25. 7. Berger M, Ure B, Lacher M. Mitomycin C in the therapy of recurrent esophageal strictures: hype or hope. Eur J Pediatr Surg 2012;22:109– 16.
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