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Invited Editorial

Cutting-edge Treatment for Benign Pediatric Esophageal Strictures, a Step Forward, More to Go

Mark, Jacob; Narkewicz, Michael R.

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Journal of Pediatric Gastroenterology and Nutrition: October 2018 - Volume 67 - Issue 4 - p 433-434
doi: 10.1097/MPG.0000000000002009
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See “Endoscopic Electrocautery Incisional Therapy as a Treatment for Refractory Benign Pediatric Esophageal Strictures” by Manfredi et al on page 464.

The study by Manfredi et al in this month's journal, highlights the difficulties in management of refractory pediatric esophageal strictures. Despite advances in endoscopic interventions that started with bougie dilation, moved to flexible balloon dilation and on to fixed sized balloon dilation, there is a subset of children who have difficult refractory esophageal strictures that require multiple endoscopic procedures with repetitive exposure to anesthesia, where dilations have a short-term success and minimal progress is made. Surgical resection carries its own morbidity for operative recovery and the potential for recurrence of esophageal strictures at the surgical anastomosis (1). Adjunctive therapies to supplement dilation have been attempted including steroid injection (2), mitomycin C application (3), and fully covered self-expanding metal stents (4). These techniques, however, have their own limitations and are not universally available or successful. Thus, there is a need for more effective endoscopic therapies. Manfredi et al present the largest pediatric series in the literature to date evaluating endoscopic electrocautery incisional therapy (EIT) as treatment for esophageal strictures. The authors have taken a systematic approach to a rare problem and have advanced our understanding of this newer therapeutic option. As with any study of new endoscopic treatments in children, there are strengths and potential applications from this study and limitations to the conclusions.

This study highlights the difficulty both defining refractory esophageal strictures and treatment success in pediatrics as there are no consensus definitions. In this study, refractory anastomotic strictures were defined by persistent dysphagia and failure to reach an age-appropriate esophageal lumen diameter with 5 dilations sessions within 5 months OR ≥ 7 dilation sessions during their life. The authors selected this definition based on a similar definition in adult studies (5,6). This proposed definition sets the stage for epidemiologic studies across multiple centers to help our field refine a uniform definition of refractory esophageal strictures. The technique for EIT is well defined in the study. Although technically challenging, direct visualization offers a more directed approach to incisional therapy. Using EIT, with balloon dilation in a large group of patients with refractory strictures (all due to esophageal atresia), the median number of dilation sessions decreased from 8 before EIT to 2 within the next 2 years. They also found a 61% rate of treatment success at 2 years after EIT, defined by no need for surgical resection, and <7 dilation sessions required after EIT to maintain appropriate esophageal diameter. Although these definitions for refractory strictures and treatment success fall short of the elimination of the need for dilation, taken together these results suggest that for many patients with refractory esophageal strictures, EIT may be an effective treatment modality to decrease the frequency of esophageal dilation. This advance should have a meaningful impact on the patients and their families. We agree with the authors that for patients with amenable refractory esophageal strictures, EIT should be considered and discussed with the patient and their family before committing to surgical resection.

The paper also highlights some limitations of EIT. All patients in the series had esophageal atresia repairs and anastomotic strictures. The needle knives vary by manufacturer, but are generally 5 mm cutting length and require a 2.8-mm working channel. These knives are appropriate to consider for a <1 cm stricture (7). They would be very difficult and likely more dangerous to use in long or angulated strictures. The authors report 7 adverse events (5.3%) of esophageal leaks, 3 of which were free perforations and 4 were contained fluid leaks. This highlights that EIT carries a risk of perforation similar or slightly higher than balloon dilation even when performed by an experienced provider for appropriate strictures. This is, however, balanced by improved success and outcomes. The risks and limitations should be discussed with the patient and family before the procedure, and EIT should only be performed by an endoscopist with appropriate expertise. The availability of pediatric therapeutic endoscopists trained in this modality will limit the application of this technique in many parts of the Unites States and other countries until experience with this technique expands.

In this study, EIT was also used on 22 patients with non-refractory strictures and the authors found 100% treatment success. It is, however, not known if these patients would have responded to alternative methods such as adjunct therapies (steroids, mitomycin C), or stenting. One randomized controlled trial in adults with naïve anastomotic esophageal strictures found no difference in mean number of dilations or treatment success between patients who underwent EIT versus bougie dilations (8). Given the technical limitations and potential increased risk of EIT, its clinical use should be limited to short refractory esophageal strictures in children unless future research shows clear benefit in other situations. In addition, we encourage the community of pediatric interventional endoscopists to work toward uniform definitions for strictures and additional outcome measures including patient-reported outcomes and systematic approaches to classifications of complications (9) as investigations in this field continue.

REFERENCES

1. Honkoop P, Siersema PD, Tilanus HW, et al. Benign anastomotic strictures after transhiatal esophagectomy and cervical esophagogastrostomy: risk factors and management. J Thorac Cardiovasc Surg 1996; 111:1141–1146. discussion 7-8.
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6. Kochman ML, McClave SA, Boyce HW. The refractory and the recurrent esophageal stricture: a definition. Gastrointest Endosc 2005; 62:474-5.
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8. Hordijk ML, van Hooft JE, Hansen BE, et al. A randomized comparison of electrocautery incision with Savary bougienage for relief of anastomotic gastroesophageal strictures. Gastrointest Endosc 2009; 70:849–855.
9. Kramer RE, Narkewicz MR. Adverse events following gastrointestinal endoscopy in children: classifications, characerizations, and implications. J Pediatr Gastroenterol Nutr 2016; 62:828–833.
Copyright © 2018 by European Society for Pediatric Gastroenterology, Hepatology, and Nutrition and North American Society for Pediatric Gastroenterology, Hepatology, and Nutrition