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Chen H, Chen ZX, Shi GQ. Risk factors and prevention and treatment methods of anastomotic stricture after esophageal atresia repair: a literature review. Pediatr Surg Int 2025; 41:99. [PMID: 40126725 DOI: 10.1007/s00383-025-05996-x] [Citation(s) in RCA: 0] [Impact Index Per Article: 0] [Reference Citation Analysis] [Abstract] [Key Words] [MESH Headings] [Journal Information] [Submit a Manuscript] [Subscribe] [Scholar Register] [Accepted: 03/06/2025] [Indexed: 03/26/2025]
Abstract
This review examines the common risk factors associated with anastomotic stricture (AS) following esophageal atresia (EA) repair and evaluates the current understanding and clinical practices concerning esophageal dilation, which remains the predominant therapeutic approach. We present emerging evidence on adjunctive therapies for recurrent and refractory strictures, including esophageal stenting, endoscopic incision therapy, mitomycin C application, local steroid injections, cell sheet transplantation, and surgical interventions. A comprehensive literature review was conducted using PubMed, with a cutoff date of December 31, 2024, encompassing all relevant studies and reviews pertinent to this topic, with a particular emphasis on refractory and recurrent strictures. Given the scarcity of evidence-based data on AS in children with EA, we also critically analyze findings from adult literature and studies addressing esophageal strictures caused by various etiologies to provide a more comprehensive understanding. The pathogenesis of AS is multifactorial, with a key contributing factor being the significant gap between the proximal and distal esophageal segments. This anatomical disparity increases anastomotic tension, thereby elevating the likelihood of stricture formation. A thorough identification and detailed understanding of the risk factors associated with AS are crucial for enhancing patient outcomes and developing effective preventive strategies. Currently, the evidence regarding esophageal dilation is insufficient to establish the superiority of any single technique, particularly between balloon and bougie dilators. Recurrent and refractory strictures remain a challenge, as no pharmacological and mechanical adjunctive interventions have demonstrated sufficient feasibility, efficacy, and safety to fundamentally change clinical practice. While some results show promise, transformative outcomes have not yet been achieved, and further research is needed to establish evidence-based best treatment strategies.
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Affiliation(s)
- Hao Chen
- Department of Gastroenterology, Hospital for Digestive Diseases, Affiliated Hospital of Zunyi Medical University, No. 149 Dalian Road, Zunyi, 563000, Guizhou, China
| | - Zhu-Xin Chen
- Department of Gastroenterology, Hospital for Digestive Diseases, Affiliated Hospital of Zunyi Medical University, No. 149 Dalian Road, Zunyi, 563000, Guizhou, China
| | - Guo-Qing Shi
- Department of Gastroenterology, Hospital for Digestive Diseases, Affiliated Hospital of Zunyi Medical University, No. 149 Dalian Road, Zunyi, 563000, Guizhou, China.
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Walker H, Salim A, Smith C, Murphy F. Developing balloon dilatation sizing guidance for anastomotic stricture dilatation following oesophageal atresia repair. Pediatr Surg Int 2023; 39:252. [PMID: 37624471 DOI: 10.1007/s00383-023-05536-5] [Citation(s) in RCA: 0] [Impact Index Per Article: 0] [Reference Citation Analysis] [Abstract] [Key Words] [MESH Headings] [Track Full Text] [Journal Information] [Submit a Manuscript] [Subscribe] [Scholar Register] [Accepted: 08/07/2023] [Indexed: 08/26/2023]
Abstract
PURPOSE To develop guidance for the selection of balloon catheter size when performing an oesophageal dilatation for a stricture post oesophageal atresia repair. METHODS This was a single centre retrospective study at a paediatric tertiary centre. Dilatations were performed between 2015 and 2020. All dilatations were performed under general anaesthesia using balloon catheters under fluoroscopic guidance. Outliers were excluded using ROUT method and descriptive analysis was calculated to 1SD or IQR depending on the normality of data distribution. RESULTS 97 patients underwent 341 dilatations. Median age was 19 months (37 weeks corrected gestation-17 years), median weight was 10.7 kg (2.6-72 kg). Balloon catheter sizes ranged from 6-8 mm to 18-20 mm. There was strong correlation between weight and balloon size (r = 0.8, p < 0.0001). There were 2 perforations (0.6%), both diagnosed intra-operatively and treated conservatively. From the results, weight recommendations were created for each balloon size. CONCLUSION Fluoroscopic balloon dilatation is a safe and effective method to treat anastomotic stricture following oesophageal atresia repair. Previous studies have shown correlation between patient weight and oesophageal diameter. We propose guidance for using an appropriate balloon size based on patient's weight with the aim to reduce complication.
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Affiliation(s)
- Hamish Walker
- Department of Paediatric Surgery, Alder Hey Children's NHS Foundation Trust, Eaton Rd, Liverpool, L12 2AP, UK.
| | - Adeline Salim
- Department of Paediatric Surgery, Alder Hey Children's NHS Foundation Trust, Eaton Rd, Liverpool, L12 2AP, UK
| | - Charlotte Smith
- Department of Paediatric Surgery, Alder Hey Children's NHS Foundation Trust, Eaton Rd, Liverpool, L12 2AP, UK
| | - Fiona Murphy
- Department of Paediatric Surgery, Alder Hey Children's NHS Foundation Trust, Eaton Rd, Liverpool, L12 2AP, UK
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Abstract
Esophageal dilations in children are performed by several pediatric and adult professionals. We aim to summarize improvements in safety and new technology used for the treatment of complex and refractory strictures, including triamcinolone injection, endoscopic electro-incisional therapy, topical mitomycin-C application, stent placement, functional lumen imaging probe assisted dilation, and endoscopic vacuum-assisted closure in the pediatric population.
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Liu S, Fang Y, Lv Y, Zhao J, Luo R, Luo R, Cheng J, Yang H, Zhang A, Shen Y, Jiang N. Magnetic compression stricturoplasty in patients with severe stricture after simultaneous esophageal atresia and duodenal obstruction repair: A case report. Exp Ther Med 2022; 23:93. [PMID: 34976135 PMCID: PMC8674971 DOI: 10.3892/etm.2021.11016] [Citation(s) in RCA: 1] [Impact Index Per Article: 0.3] [Reference Citation Analysis] [Abstract] [Key Words] [Track Full Text] [Download PDF] [Figures] [Journal Information] [Subscribe] [Scholar Register] [Received: 03/01/2020] [Accepted: 10/07/2021] [Indexed: 01/27/2023] Open
Abstract
Combined esophageal atresia (EA), tracheoesophageal fistula (TEF) and duodenal obstruction result in various challenges in management, and a well-defined management protocol is still lacking. Esophageal stricture is the most common complication after EA repair. The use of magnetic compression alimentary tract anastomosis has been reported in children. By searching the literature, the present study reports the first case of simultaneous repair (EA repair followed by duodenal obstruction repair) and magnetic compression stricturoplasty for refractory esophageal stricture after EA repair in two male neonates. One of the neonates received delayed treatment of duodenal obstruction, and the other successfully underwent a simultaneous emergency operation of these combined anomalies. These two infants developed refractory strictures despite multiple endoscopic dilatation procedures during the postoperative follow-up period. Magnetic compression stricturoplasty procedures were successfully performed under fluoroscopic and endoscopic guidance without any leakage or complication. At the follow-up 10-months after stricturoplasty, the two patients achieved durable esophageal patency in the absence of dysphagia. Combination of early chest and abdominal X-ray detection is recommended to avoid a delayed diagnosis and treatment, as well as the synchronous operation for EA/TEF repair and duodenoduodenostomy in a single surgery for combined EA/TEF and duodenal obstructions. Therefore, magnetic compression stricturoplasty is a feasible and efficient method for establishing early patency of the esophagus in patients with refractory EA stricture.
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Affiliation(s)
- Shiqi Liu
- Department of Nursing, Medical College, Xijing University, Xi'an, Shaanxi 710123, P.R. China
| | - Ying Fang
- Department of Digestive Diseases, Xi'an Children's Hospital, Xi'an, Shaanxi 710003, P.R. China
| | - Yi Lv
- Department of Hepatobiliary Surgery, The First Affiliated Hospital of Xi'an Jiaotong University, Xi'an, Shaanxi 710061, P.R. China
| | - Jingru Zhao
- Department of Pediatrics, Northwest Women's and Children's Hospital, Xi'an, Shaanxi 710061, P.R. China
| | - Ruixue Luo
- Corrosion and Protection Research Lab (CPRL), Northwest Institute for Nonferrous Metal Research, Xi'an, Shaanxi 710016, P.R. China
| | - Ruogu Luo
- Department of Pediatrics, Northwest Women's and Children's Hospital, Xi'an, Shaanxi 710061, P.R. China
| | - Jun Cheng
- Pediatric Intensive Care Unit, Northwest Women's and Children's Hospital, Xi'an, Shaanxi 710061, P.R. China
| | - Hongbin Yang
- Department of Digestive Diseases, Xi'an Children's Hospital, Xi'an, Shaanxi 710003, P.R. China
| | - Anpeng Zhang
- Department of Pediatrics, Northwest Women's and Children's Hospital, Xi'an, Shaanxi 710061, P.R. China
| | - Yingchun Shen
- Pediatric Intensive Care Unit, Northwest Women's and Children's Hospital, Xi'an, Shaanxi 710061, P.R. China
| | - Na Jiang
- Department of Anesthesia, Northwest Women's and Children's Hospital, Xi'an, Shaanxi 710061, P.R. China
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van Hal ARL, Pulvirenti R, den Hartog FPJ, Vlot J. The Safety of Intralesional Steroid Injections in Young Children and Their Effectiveness in Anastomotic Esophageal Strictures-A Meta-Analysis and Systematic Review. Front Pediatr 2021; 9:825030. [PMID: 35165653 PMCID: PMC8837747 DOI: 10.3389/fped.2021.825030] [Citation(s) in RCA: 3] [Impact Index Per Article: 0.8] [Reference Citation Analysis] [Abstract] [Key Words] [Track Full Text] [Download PDF] [Figures] [Journal Information] [Submit a Manuscript] [Subscribe] [Scholar Register] [Received: 11/29/2021] [Accepted: 12/27/2021] [Indexed: 12/03/2022] Open
Abstract
OBJECTIVE Intralesional steroid injections (ISI) are a widely used technique for various pediatric indications and represent a possible adjuvant treatment for anastomotic esophageal strictures. Yet, no consensus has been reached neither on their safety in the pediatric population or their effectiveness in esophageal atresia patients. This systematic review aimed to assess the safety of ISI in young children through a meta-analysis and to summarize the current knowledge on the effectiveness of ISI in anastomotic esophageal strictures. METHODS A systematic literature search was performed in Embase, Medline, Web of Science Core Collection, Cochrane Central Register of Controlled Trials and Google Scholar up to August 16 2021. Studies focusing on ISI and involving children up to 2 years were included in the meta-analysis for the safety assessment. All studies evaluating the use of ISI as adjuvant treatment in anastomotic esophageal strictures in children were included in the systematic review to assess the effectiveness of the intervention. RESULTS The literature search yielded 8,253 articles. A total of 57 studies were included, of which 55 for the safety and five for the effectiveness assessment. The overall complication rate was 7%, with a greater incidence of local complications compared to systemic complications. Six studies (with a total of 367 patients) evaluated adrenocorticotropic hormone and cortisol levels, of which four reported hypothalamic-pituitary axis suppression. Two children (0.6%) received replacement therapy and all patients recovered uneventfully. A mean number of 1.67 ISI were performed per esophageal atresia (EA) patient. A reduction of needed dilatations was seen after ISI, compared to the number of dilatations performed before the intervention (5.2 vs. 1.3). CONCLUSION The insufficient data emphasized the need for further prospective and comparative studies. Results from this meta-analysis and systematic review address ISI as a safe and effective technique. Close clinical follow-up and growth curve evaluation are advisable in patients receiving ISI. SYSTEMATIC REVIEW REGISTRATION PROSPERO, identifier: CRD42021281584.
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Affiliation(s)
- Annefleur R L van Hal
- Department of Pediatric Surgery and Intensive Care, Erasmus Medical Centre Sophia Children's Hospital, Rotterdam, Netherlands
| | - Rebecca Pulvirenti
- Pediatric Surgery Unit, Department of Women's and Children's Health, University Hospital of Padua, Padua, Italy
| | | | - John Vlot
- Department of Pediatric Surgery and Intensive Care, Erasmus Medical Centre Sophia Children's Hospital, Rotterdam, Netherlands
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Intralesional Steroid Injection Therapy for Esophageal Anastomotic Stricture Following Esophageal Atresia Repair. J Pediatr Gastroenterol Nutr 2020; 70:462-467. [PMID: 31764412 DOI: 10.1097/mpg.0000000000002562] [Citation(s) in RCA: 19] [Impact Index Per Article: 3.8] [Reference Citation Analysis] [Abstract] [Track Full Text] [Journal Information] [Submit a Manuscript] [Subscribe] [Scholar Register] [Indexed: 12/13/2022]
Abstract
OBJECTIVES The role of intralesional steroid injection (ISI) in the treatment of anastomotic stricture in patients with esophageal atresia remains unclear. The aim of this study was to evaluate the efficacy and safety of ISI. METHODS A total of 158 patients with esophageal atresia with at least 1 ISI for the treatment of esophageal anastomotic stricture between 2010 and 2017 were identified. The change in stricture diameter (ΔD) was compared between procedures with dilation alone (ISI-) and dilation with steroid injection (ISI+). RESULTS A total of 1055 balloon dilations were performed (452 ISI+). The median ΔD was significantly greater in the ISI+ group: 1 mm (interquartile range [IQR] 0, 3) versus 0 mm (IQR -1, 1.5) (P < 0.0001). The ISI+ group had greater percentage of improved diameter (P < 0.0001) and lesser percentages of unchanged and decreased diameters at subsequent endoscopy (P = 0.0009, P = 0.003). Multivariable logistic regression confirmed the significance of ISI on increasing the likelihood of improved stricture diameter with an adjusted odds ratio of 3.24 (95% confidence interval: 2.15-4.88) (P < 0.001). The ΔD for the first 3 ISI+ procedures was greater than the ΔD for subsequent ISI+ procedures: 1 mm (IQR 0, 3) versus 0.5 mm (IQR-1.25, 2) (P = 0.001). There was no difference in perforation incidence between ISI+ and ISI- groups (P = 0.82). CONCLUSIONS ISI with dilation was well tolerated and improved anastomotic stricture diameter more than dilation alone. The benefit of ISI over dilation alone was limited to the first 3 ISI procedures.
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Lange B, Sold M, Kähler G, Wessel LM, Kubiak R. Experience with fully covered self-expandable metal stents for anastomotic stricture following esophageal atresia repair. Dis Esophagus 2018; 31:5043492. [PMID: 29939253 DOI: 10.1093/dote/doy061] [Citation(s) in RCA: 10] [Impact Index Per Article: 1.4] [Reference Citation Analysis] [Abstract] [Track Full Text] [Journal Information] [Submit a Manuscript] [Subscribe] [Scholar Register] [Received: 11/09/2017] [Accepted: 06/08/2018] [Indexed: 12/11/2022]
Abstract
There is a lack of experience with fully covered self-expandable metal stents (SEMSs) for the treatment of benign esophageal conditions in the pediatric population. This is the evaluation of our institutional experience of placing SEMSs for anastomotic stricture (AS) formation following esophageal atresia (EA) repair. Patients were jointly managed from the Department of Pediatric Surgery and Central Interdisciplinary Endoscopy at our institution. Thirteen children (8 male, 5 female) with a median age of 4 months (range: 1-32 months) who underwent treatment with SEMSs for a postoperative AS following EA repair between February 2006 and April 2016 were recruited into this retrospective study. SEMSs that are originally designed for other organs such as trachea, bronchus, biliary tract, or colon were inserted under general anesthesia via endoscopic guidance. Simultaneous fluoroscopy was not required in any case. In five infants, the stents were inserted primarily without previous therapy. Seven patients underwent stenting following dilatation with or without adjuncts (e.g. Mitomycin C, Triamcinolone). In one case with an AS and a simultaneous persistent tracheoesophageal fistula (TEF), multiple SEMSs were applied after failure to close the fistula with fibrin glue.The median duration of individual stent placement was 30 days (range: 5-91 days). In five children up to four different biliary, bronchial or colonic SEMSs were placed successively over time. There were no problems noted at stent insertion or removal. Eight children (62%) developed complications associated with stenting. At follow-up, in eight patients (62%) AS was resolved, including all of those five cases, who had their stents inserted without previous therapy. Five children (38%), who underwent dilatation prior to stenting did not improve their AS and required further intervention. Overall, the cohort exhibited a slight, but not significant weight gain between stent insertion and (final) stent removal.Insertion of SEMSs for AS following EA repair is safe and often successful with only one single application. It can be used as a primary procedure (without previous therapy) or after failed dilatations.There was one death in this study that was unrelated to stenting and occurred 12 months after stent removal. Because of the absence of manufactured, age-related devices, SEMSs that are originally designed for other organs can be applied. Establishment of a standardized management including stent placement for the treatment of AS following EA repair in the pediatric population is required.
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Affiliation(s)
- B Lange
- Department of Pediatric Surgery, Heidelberg University, Theodor-Kutzer-Ufer, Mannheim, Germany
| | - M Sold
- Central Interdisciplinary Endoscopy, University Medical Center Mannheim, Medical Faculty Mannheim, Heidelberg University, Theodor-Kutzer-Ufer, Mannheim, Germany
| | - G Kähler
- Central Interdisciplinary Endoscopy, University Medical Center Mannheim, Medical Faculty Mannheim, Heidelberg University, Theodor-Kutzer-Ufer, Mannheim, Germany
| | - L M Wessel
- Department of Pediatric Surgery, Heidelberg University, Theodor-Kutzer-Ufer, Mannheim, Germany
| | - R Kubiak
- Department of Pediatric Surgery, Heidelberg University, Theodor-Kutzer-Ufer, Mannheim, Germany
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Alonso V, Ojha D, Nalluri H, de Agustín JC. Use of self-expanding nitinol stents in the pediatric management of refractory esophageal caustic stenosis. REVISTA ESPANOLA DE ENFERMEDADES DIGESTIVAS 2017; 109:728-730. [PMID: 28942656 DOI: 10.17235/reed.2017.4959/2017] [Citation(s) in RCA: 1] [Impact Index Per Article: 0.1] [Reference Citation Analysis] [Abstract] [Track Full Text] [Subscribe] [Scholar Register] [Indexed: 11/20/2022]
Abstract
BACKGROUND The treatment of recurrent esophageal stricture secondary to the ingestion of a caustic agent is an arduous task. Self-expanding esophageal stents may be an alternative to repeated endoscopic esophageal dilations. CASE REPORT We present the case of a two-year-old male with a severe and long esophageal stricture successfully treated by the combination of dilations and stent placement. After five months of serial pneumatic dilations, three self-expanding nitinol stents internally coated with silicone were introduced through a gastrostomy, covering the entire esophagus. The procedure was performed under endoscopic and radiological guidance. Three months later, the treatment was repeated with a single stent. A new stenosis in the proximal esophagus required surgical resection, and anastomosis followed by two pneumatic dilations for five months resulted in longer intervals where the patient was asymptomatic. DISCUSSION The results obtained were satisfactory, allowing the patient to conserve and use his own esophagus. However, this is a unique case and the optimal maintenance time and withdrawal time of the stent must be determined.
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Affiliation(s)
- Verónica Alonso
- Cirugía Pediátrica, Hospital Clínico Universitario de Valladolid, España
| | - Devicka Ojha
- Internal Medicine Department, Ohio State University Hospitals (USA), USA
| | - Harsha Nalluri
- Radiology Department, University of Cincinnati Medical Center, Ohio (USA), USA
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Woo R, Wong CM, Trimble Z, Puapong D, Koehler S, Miller S, Johnson S. Magnetic Compression Stricturoplasty For Treatment of Refractory Esophageal Strictures in Children: Technique and Lessons Learned. Surg Innov 2017; 24:432-439. [PMID: 28745145 DOI: 10.1177/1553350617720994] [Citation(s) in RCA: 21] [Impact Index Per Article: 2.6] [Reference Citation Analysis] [Abstract] [Key Words] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Indexed: 12/11/2022]
Abstract
INTRODUCTION Esophageal stricture is the most common complication following repair of esophageal atresia (EA). In general, these strictures are successfully managed using endoscopic techniques including bougie and balloon dilation, stenting, and chemotherapeutic agent application. If these techniques are unsuccessful, patients require segmental esophageal resection and reanastomosis or esophageal replacement. Magnetic compression anastomosis has been described in children. Herein we report our experience with magnetic compression stricturoplasty to treat refractory strictures after EA repair. METHODS We reviewed our experience using magnets to treat refractory strictures in 2 patients. Both patients failed multiple standard interventions. Because of near complete esophageal obstruction, both patients were candidates for esophageal replacement or segmental resection/anastamosis. In both patients, we applied neodymium-iron-boron magnets using fluoroscopic and endoscopic guidance. RESULTS The magnets were successfully positioned in both cases. Magnets were left in place for 7 and 10 days allowing for gradual compression stricturoplasty/anastamosis. Upon removal of the magnets, recanalization was visualized endoscopically and self-expanding stents were placed. There were no leaks or significant early complications. By 31 months post-magnetic stricturoplasty, both patients achieved durable esophageal patency without dysphagia. CONCLUSION Magnetic stricturoplasty was successful at establishing early patency of the esophagus in 2 patients with recalcitrant EA strictures. Fundamental knowledge of magnetism was critical in configuring magnet arrays for surgery. In both cases, early follow-up is promising. Further follow-up will define the long-term success of this technique.
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Tambucci R, Angelino G, De Angelis P, Torroni F, Caldaro T, Balassone V, Contini AC, Romeo E, Rea F, Faraci S, Federici di Abriola G, Dall'Oglio L. Anastomotic Strictures after Esophageal Atresia Repair: Incidence, Investigations, and Management, Including Treatment of Refractory and Recurrent Strictures. Front Pediatr 2017; 5:120. [PMID: 28611969 PMCID: PMC5447026 DOI: 10.3389/fped.2017.00120] [Citation(s) in RCA: 60] [Impact Index Per Article: 7.5] [Reference Citation Analysis] [Abstract] [Key Words] [Track Full Text] [Download PDF] [Figures] [Journal Information] [Submit a Manuscript] [Subscribe] [Scholar Register] [Received: 01/25/2017] [Accepted: 05/04/2017] [Indexed: 01/10/2023] Open
Abstract
Improved surgical techniques, as well as preoperative and postoperative care, have dramatically changed survival of children with esophageal atresia (EA) over the last decades. Nowadays, we are increasingly seeing EA patients experiencing significant short- and long-term gastrointestinal morbidities. Anastomotic stricture (AS) is the most common complication following operative repair. An esophageal stricture is defined as an intrinsic luminal narrowing in a clinically symptomatic patient, but no symptoms are sensitive or specific enough to diagnose an AS. This review aims to provide a comprehensive view of AS in EA children. Given the lack of evidence-based data, we critically analyzed significant studies on children and adults, including comments on benign strictures with other etiologies. Despite there is no consensus about the goal of the luminal diameter based on the patient's age, esophageal contrast study, and/or endoscopy are recommended to assess the degree of the narrowing. A high variability in incidence of ASs is reported in literature, depending on different definitions of AS and on a great number of pre-, intra-, and postoperative risk factor influencing the anastomosis outcome. The presence of a long gap between the two esophageal ends, with consequent anastomotic tension, is determinant for stricture formation and its response to treatment. The cornerstone of treatment is endoscopic dilation, whose primary aims are to achieve symptom relief, allow age-appropriate capacity for oral feeding, and reduce the risk of pulmonary aspiration. No clear advantage of either balloon or bougie dilator has been demonstrated; therefore, the choice is based on operator experience and comfort with the equipment. Retrospective evidences suggest that selective dilatations (performed only in symptomatic patients) results in significantly less number of dilatation sessions than routine dilations (performed to prevent symptoms) with equal long-term outcomes. The response to dilation treatment is variable, and some patients may experience recurrent and refractory ASs. Adjunctive treatments have been used, including local injection of steroids, topical application of mitomycin C, and esophageal stenting, but long-term studies are needed to prove their efficacy and safety. Stricture resection or esophageal replacement with an interposition graft remains options for AS refractory to conservative treatments.
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Affiliation(s)
- Renato Tambucci
- Digestive Endoscopy and Surgery Unit, Bambino Gesù Children's Hospital, IRCCS, Rome, Italy.,University of L'Aquila, L'Aquila, Italy
| | - Giulia Angelino
- Digestive Endoscopy and Surgery Unit, Bambino Gesù Children's Hospital, IRCCS, Rome, Italy
| | - Paola De Angelis
- Digestive Endoscopy and Surgery Unit, Bambino Gesù Children's Hospital, IRCCS, Rome, Italy
| | - Filippo Torroni
- Digestive Endoscopy and Surgery Unit, Bambino Gesù Children's Hospital, IRCCS, Rome, Italy
| | - Tamara Caldaro
- Digestive Endoscopy and Surgery Unit, Bambino Gesù Children's Hospital, IRCCS, Rome, Italy
| | - Valerio Balassone
- Digestive Endoscopy and Surgery Unit, Bambino Gesù Children's Hospital, IRCCS, Rome, Italy
| | - Anna Chiara Contini
- Digestive Endoscopy and Surgery Unit, Bambino Gesù Children's Hospital, IRCCS, Rome, Italy
| | - Erminia Romeo
- Digestive Endoscopy and Surgery Unit, Bambino Gesù Children's Hospital, IRCCS, Rome, Italy
| | - Francesca Rea
- Digestive Endoscopy and Surgery Unit, Bambino Gesù Children's Hospital, IRCCS, Rome, Italy
| | - Simona Faraci
- Digestive Endoscopy and Surgery Unit, Bambino Gesù Children's Hospital, IRCCS, Rome, Italy
| | | | - Luigi Dall'Oglio
- Digestive Endoscopy and Surgery Unit, Bambino Gesù Children's Hospital, IRCCS, Rome, Italy
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ESPGHAN-NASPGHAN Guidelines for the Evaluation and Treatment of Gastrointestinal and Nutritional Complications in Children With Esophageal Atresia-Tracheoesophageal Fistula. J Pediatr Gastroenterol Nutr 2016; 63:550-570. [PMID: 27579697 DOI: 10.1097/mpg.0000000000001401] [Citation(s) in RCA: 240] [Impact Index Per Article: 26.7] [Reference Citation Analysis] [Abstract] [Track Full Text] [Journal Information] [Submit a Manuscript] [Subscribe] [Scholar Register] [Indexed: 12/17/2022]
Abstract
BACKGROUND Esophageal atresia (EA) is one of the most common congenital digestive anomalies. With improvements in surgical techniques and intensive care treatments, the focus of care of these patients has shifted from mortality to morbidity and quality-of-life issues. These children face gastrointestinal (GI) problems not only in early childhood but also through adolescence and adulthood. There is, however, currently a lack of a systematic approach to the care of these patients. The GI working group of International Network on Esophageal Atresia comprises members from ESPGHAN/NASPGHAN and was charged with the task of developing uniform evidence-based guidelines for the management of GI complications in children with EA. METHODS Thirty-six clinical questions addressing the diagnosis, treatment, and prognosis of the common GI complications in patients with EA were formulated. Questions on the diagnosis, and treatment of gastroesophageal reflux, management of "cyanotic spells," etiology, investigation and management of dysphagia, feeding difficulties, anastomotic strictures, congenital esophageal stenosis in EA patients were addressed. The importance of excluding eosinophilic esophagitis and associated GI anomalies in symptomatic patients with EA is discussed as is the quality of life of these patients and the importance of a systematic transition of care to adulthood. A systematic literature search was performed from inception to March 2014 using Embase, MEDLINE, the Cochrane Database of Systematic Reviews, Cochrane Central Register of Controlled Clinical Trials, and PsychInfo databases. The approach of the Grading of Recommendations Assessment, Development and Evaluation was applied to evaluate outcomes. During 2 consensus meetings, all recommendations were discussed and finalized. The group members voted on each recommendation, using the nominal voting technique. Expert opinion was used where no randomized controlled trials were available to support the recommendation.
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12
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Manfredi MA. Endoscopic Management of Anastomotic Esophageal Strictures Secondary to Esophageal Atresia. Gastrointest Endosc Clin N Am 2016; 26:201-19. [PMID: 26616905 DOI: 10.1016/j.giec.2015.09.002] [Citation(s) in RCA: 30] [Impact Index Per Article: 3.3] [Reference Citation Analysis] [Abstract] [Key Words] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Indexed: 02/07/2023]
Abstract
The reported incidence of anastomotic stricture after esophageal atresia repair has varied in case series from as low as 9% to as high as 80%. The cornerstone of esophageal stricture treatment is dilation with either balloon or bougie. The goal of esophageal dilation is to increase the luminal diameter of the esophagus while also improving dysphagia symptoms. Once a stricture becomes refractory to esophageal dilation, there are several treatment therapies available as adjuncts to dilation therapy. These therapies include intralesional steroid injection, mitomycin C, esophageal stent placement, and endoscopic incisional therapy.
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Affiliation(s)
- Michael A Manfredi
- Esophageal and Airway Atresia Treatment Center, Boston Children's Hospital, Boston, MA 02132, USA; Pediatrics Harvard Medical School, Boston, MA 02115, USA.
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13
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Bouguermouh D, Salem A. Esophageal atresia: a critical review of management at a single center in Algeria. Dis Esophagus 2015; 28:205-10. [PMID: 24467412 DOI: 10.1111/dote.12174] [Citation(s) in RCA: 9] [Impact Index Per Article: 0.9] [Reference Citation Analysis] [Abstract] [Key Words] [Track Full Text] [Journal Information] [Submit a Manuscript] [Subscribe] [Scholar Register] [Indexed: 12/11/2022]
Abstract
The purpose was to study the outcomes and factors affecting the survival of esophageal atresia in our center. A retrospective analysis of 86 cases of esophageal atresia (EA) over a 10-year period was performed with 46 boys and 42 girls. Demographic data, birth weight, gestational age, consanguinity, incidence of associated anomalies, place of delivery, history of feeding, and outcomes were studied. EA with distal tracheoesophageal fistula (TEF) was the commonest type with 58/86 (67%). The percentage of patients with at least one associated anomaly was 52/86 (60%), with 7/86 (8%) who are from consanguineous parents; most commonly associated anomalies were cardiac 13/86 (15%). The average gestational age and birth weight were 36 ± 2 weeks and 2300 ± 570 g, respectively. Survival rates for the patients according to the Waterston classification was 80% in group A, 58% in group B, and 25% in group C (three patients died before surgery). Prematurity, the gap between the two ends of the esophagus, and preoperative respiratory status were the most significant factors affecting the survival. Late complication of EA/TEF include respiratory symptoms, especially in the first year, associating tracheomalacia and bronchopulmonary infections in about 24/45 (53%), recurrence of TEF 3/45 (7%), esophageal stricture 26/45 (58%), and gastroesophageal reflux 22/45 (49%). The high incidence of delayed diagnosis, low birth weight, and lack of advanced neonatological management are important contributory factors to the poor outcome. The frequency of late complications highlights the need for multidisciplinary clinics to follow these children's.
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Affiliation(s)
- D Bouguermouh
- Pediatric Surgery Department, Pr Nefissa Hamoud University Hospital Center, Algiers, Algeria; Faculty of Medical Sciences, University of Algiers, Algiers, Algeria
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Sun LYC, Laberge JM, Yousef Y, Baird R. The Esophageal Anastomotic Stricture Index (EASI) for the management of esophageal atresia. J Pediatr Surg 2015; 50:107-10. [PMID: 25598104 DOI: 10.1016/j.jpedsurg.2014.10.008] [Citation(s) in RCA: 18] [Impact Index Per Article: 1.8] [Reference Citation Analysis] [Abstract] [Key Words] [Track Full Text] [Journal Information] [Submit a Manuscript] [Subscribe] [Scholar Register] [Received: 09/28/2014] [Accepted: 10/06/2014] [Indexed: 12/26/2022]
Abstract
BACKGROUND Anastomotic stricture is the most common complication following repair of esophageal atresia. An Esophageal Anastomotic Stricture Index (EASI) based on the postoperative esophagram may identify patients at high risk of stricture formation. METHODS Digital images of early postoperative esophagrams of patients undergoing EA repair from 2005 to 2013 were assessed. Demographics and outcomes including dilations were prospectively collected. Upper (U-EASI) and lower (L-EASI) pouch ratios were generated using stricture diameter divided by maximal respective pouch diameter. Score performances were evaluated with area under the receiver operator curves (AUC) and the Fisher's exact test for single and multiple (>3) dilatations. Interrater agreement was evaluated using the intraclass correlation coefficient (ICC). RESULTS Forty-five patients had esophagrams analyzed; 28 (62%) required dilatation and 19 received >3 (42%). U-EASI and L-EASI ratios ranged from 0.17 to 0.70, with L-EASI outperforming the U-EASI as follows: L-EASI AUC: 0.66 for a single dilatation, 0.65 for >3 dilatations; U-EASI AUC: 0.56 for a single dilatation, 0.67 for >3 dilatations. All patients with an L-EASI ratio of ≤0.30 (n=8) required multiple esophageal dilatations, p=0.0006. The interrater ICC was 0.87. CONCLUSION The EASI is a simple, reproducible tool to predict the development and severity of anastomotic stricture after esophageal atresia repair and can direct postoperative surveillance.
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Affiliation(s)
| | - Jean-Martin Laberge
- Division of Pediatric Surgery, Montreal Children's Hospital; Faculty of Medicine, McGill University, Montreal, QC, Canada
| | | | - Robert Baird
- Division of Pediatric Surgery, Montreal Children's Hospital; Faculty of Medicine, McGill University, Montreal, QC, Canada.
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Kassabian S, Baez-Socorro V, Sferra T, Garcia R. Eosinophilic esophagitis in patients with esophageal atresia and chronic dysphagia. World J Gastroenterol 2014; 20:18038-18043. [PMID: 25548504 PMCID: PMC4273156 DOI: 10.3748/wjg.v20.i47.18038] [Citation(s) in RCA: 16] [Impact Index Per Article: 1.5] [Reference Citation Analysis] [Abstract] [Key Words] [MESH Headings] [Track Full Text] [Download PDF] [Journal Information] [Submit a Manuscript] [Subscribe] [Scholar Register] [Received: 03/13/2014] [Revised: 07/22/2014] [Accepted: 09/19/2014] [Indexed: 02/06/2023] Open
Abstract
Esophageal atresia (EA) is defined as a discontinuity of the lumen of the esophagus repaired soon after birth. Dysphagia is a common symptom in these patients, usually related to stricture, dysmotility or peptic esophagitis. We present 4 cases of patients with EA who complained of dysphagia and the diagnosis of Eosinophilic esophagitis (EoE) was made, ages ranging from 9 to 16 years. Although our patients were on acid suppression years after their EA repair, they presented with acute worsening of dysphagia. Esophogastroduodenoscopy and/or barium swallow did not show stricture and biopsies revealed elevated eosinophil counts consistent with EoE. Two of 4 patients improved symptomatically with the topical steroids. It is important to note that all our patients have asthma and 3 out of 4 have tested positive for food allergies. One of our patients developed recurrent anastomotic strictures that improved with the treatment of the EoE. A previous case report linked the recurrence of esophageal strictures in patients with EA repair with EoE. Once the EoE was treated the strictures resolved. On the other hand, based on our observation, EoE could be present in patients without recurrent anastomotic strictures. There appears to be a spectrum in the disease process. We are suggesting that EoE is a frequent concomitant problem in patients with history of congenital esophageal deformities, and for this reason any of these patients with refractory reflux symptoms or dysphagia (with or without anastomotic stricture) may benefit from an endoscopic evaluation with biopsies to rule out EoE.
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16
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Topical mitomycin-C application in recurrent esophageal strictures after surgical repair of esophageal atresia. J Pediatr Gastroenterol Nutr 2014; 59:608-11. [PMID: 24590215 DOI: 10.1097/mpg.0000000000000352] [Citation(s) in RCA: 24] [Impact Index Per Article: 2.2] [Reference Citation Analysis] [Abstract] [Track Full Text] [Journal Information] [Submit a Manuscript] [Subscribe] [Scholar Register] [Indexed: 02/07/2023]
Abstract
OBJECTIVES The aim of the present study was to evaluate the efficacy and short-term safety of topical mitomycin-C, an antifibrotic agent, in preventing the recurrence of anastomotic strictures after surgical repair of esophageal atresia (EA). METHODS We retrospectively reviewed the medical records of patients with recurrent anastomotic strictures after EA surgery who underwent at least 3 esophageal dilations. We compared the outcome (ie, resolution of the stricture) of the group that received topical mitomycin-C treatment with endoscopic esophageal dilation with a historical cohort treated by dilations alone. RESULTS A total of 11 children received mitomycin-C concurrently with endoscopic dilations. After a median follow-up of 33 months (range 18-72), and a mean number of 5.4 dilations per patient (range 3-11), 8 of 11 patients achieved a resolution of their strictures, 2 patients remained with stenosis, and 1 patient needed a surgical correction. In the control group, 10 patients required an average of 3.7 (range 3-7) total dilations. After a follow-up of 125 months (range 35-266) after the last dilation, strictures in 9 of 10 children disappeared and the remaining patient was symptom free. No dysplasia related to mitomycin-C was demonstrated. CONCLUSIONS There is no benefit in the resolution of the stricture when adding mitomycin-C treatment compared with repeated esophageal dilations alone in historical controls. Further randomized controlled studies and a short- and long-term evaluation of safety are needed.
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17
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The management of postoperative reflux in congenital esophageal atresia-tracheoesophageal fistula: a systematic review. Pediatr Surg Int 2014; 30:987-96. [PMID: 25011995 DOI: 10.1007/s00383-014-3548-0] [Citation(s) in RCA: 19] [Impact Index Per Article: 1.7] [Reference Citation Analysis] [Abstract] [Track Full Text] [Journal Information] [Submit a Manuscript] [Subscribe] [Scholar Register] [Accepted: 07/03/2014] [Indexed: 12/29/2022]
Abstract
PURPOSE Esophageal atresia (EA), with or without tracheoesophageal fistula (TEF), is associated with postoperative gastroesophageal reflux (GER). We performed a systematic review of the literature regarding routine anti-reflux medication post EA-TEF repair and its impact on postoperative GER and associated complications. METHODS A comprehensive search was conducted using MEDLINE, EMBASE, CINHAL, CENTRAL (Cochrane library) electronic databases and gray literature. Full-text screening was performed in duplicate. Included articles reported a primary diagnosis of EA-TEF, a secondary diagnosis of postoperative GER, and primary treatment of GER with anti-reflux medications. RESULTS Screening of 2,910 articles resulted in 25 articles (1,663 patients) for analysis. Most were single-center studies (92%) and retrospective (76%); there were no randomized control trials. Fifteen studies named the class of anti-reflux agent used, 3 the duration of therapy, and none either the dose prescribed or number of doses. Complications were inconsistently reported. Anti-reflux surgery was performed in 433/1,663 (26.0%) patients. Average follow-up was 53.2 months (14 studies). CONCLUSION The quality of literature regarding anti-reflux medication for GER post EA-TEF repair is poor. There are no well-outlined algorithms for anti-reflux agents, doses, or duration of therapy. Standardized protocols and reliable reporting are necessary to develop guidelines to better manage postoperative GER in EA-TEF patients.
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18
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Post-operative management of esophageal atresia-tracheoesophageal fistula and gastroesophageal reflux: a Canadian Association of Pediatric Surgeons annual meeting survey. J Pediatr Surg 2014; 49:716-9. [PMID: 24851754 DOI: 10.1016/j.jpedsurg.2014.02.052] [Citation(s) in RCA: 18] [Impact Index Per Article: 1.6] [Reference Citation Analysis] [Abstract] [Key Words] [Track Full Text] [Journal Information] [Submit a Manuscript] [Subscribe] [Scholar Register] [Received: 02/02/2014] [Accepted: 02/13/2014] [Indexed: 01/21/2023]
Abstract
BACKGROUND Esophageal atresia (EA), with or without tracheoesophageal fistula (TEF), is commonly associated with gastroesophageal reflux (GER) after surgical repair. One risk factor for anastomotic stricture is post-operative GER. This survey assessed practice patterns among attendees at the Canadian Association of Pediatric Surgeons (CAPS) annual meeting with respect to management of GER post EA-TEF repair. METHODS A pre-piloted survey was handed out and collected at the 2012 CAPS annual meeting. Data were entered and coded, and descriptive statistics were calculated. RESULTS We distributed 70 surveys, and 57 (81.4%) surveys were returned. On average, the incidence of EA-TEF is 8-10 cases per institution, per year. Anti-reflux medication is started immediately post-operatively in 74% of patients at institution of feeds (11%), or if symptoms of reflux develop (14%). Proton pump inhibitors and H2-receptor antagonists are used in approximately equal proportion. Patients are typically kept on anti-reflux medication for 3-6 months (37%) or 6-12 months (35%). CONCLUSIONS Most CAPS attendees treat postoperative GER prophylactically. However, there is no consistency in management strategy regarding which anti-reflux agent to use or for how long. A multi-centered study is required to establish a standardized protocol for the post-operative management of EA-TEF to prevent reflux and its effect on anastomotic strictures.
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Lévesque D, Baird R, Laberge JM. Refractory strictures post-esophageal atresia repair: what are the alternatives? Dis Esophagus 2013; 26:382-7. [PMID: 23679028 DOI: 10.1111/dote.12047] [Citation(s) in RCA: 36] [Impact Index Per Article: 3.0] [Reference Citation Analysis] [Abstract] [Track Full Text] [Journal Information] [Submit a Manuscript] [Subscribe] [Scholar Register] [Indexed: 12/11/2022]
Abstract
Esophageal strictures remain the most frequent complication after esophageal atresia (EA) repair despite refinements in operative techniques. With an incidence of anastomotic stricture between 8% and 49%, EA is the most frequent cause of benign esophageal stricture in children. The mainstay of treatment for esophageal stricture is dilatation with a 58-96% success rate. In order to relieve dysphagia, between 1 and 15 dilatations will be required in each EA patient with an esophageal stricture. However dilatations may lead to complications including perforation (0.1-0.4% of all esophageal benign strictures) and sociopsychological morbidity. Fifty percent of EA strictures will improve in 6 months. However, 30% will persist and require repeat dilatations. The present article explores the variety of non-surgical alternative treatments for anastomotic strictures after EA repair, focusing on triamcinolone acetonide, mitomycin C and esophageal stents. We propose an algorithm for a more standardized therapeutic approach, with the hope that an international panel of experts could meet and establish a consensus.
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Affiliation(s)
- D Lévesque
- Gastroenterology and Pediatric General Surgery Divisions, Montreal Children's Hospital, McGill University Health Center, Montreal, Quebec, Canada H3H1P3.
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Lakoma A, Fallon SC, Mathur S, Kim ES. Use of Mitomycin C for Refractory Esophageal Stricture following Tracheoesophageal Fistula Repair. European J Pediatr Surg Rep 2013; 1:24-6. [PMID: 25755944 PMCID: PMC4335951 DOI: 10.1055/s-0033-1341418] [Citation(s) in RCA: 1] [Impact Index Per Article: 0.1] [Reference Citation Analysis] [Abstract] [Key Words] [Track Full Text] [Download PDF] [Figures] [Journal Information] [Subscribe] [Scholar Register] [Received: 12/04/2012] [Accepted: 01/16/2013] [Indexed: 11/18/2022] Open
Abstract
Esophageal stricture is a well-described complication following tracheoesophageal fistula repair. Herein, we report two patients who had persistent esophageal strictures after several months of repeat balloon dilatations. Each patient was treated with a single application of topical mitomycin C in addition to esophageal dilatation, which resulted in complete resolution of the stricture.
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Affiliation(s)
- Anna Lakoma
- Division of Pediatric Surgery, Michael E. DeBakey Department of Surgery, Baylor College of Medicine, Houston, Texas, United States
| | - Sara C Fallon
- Division of Pediatric Surgery, Michael E. DeBakey Department of Surgery, Baylor College of Medicine, Houston, Texas, United States
| | - Shawn Mathur
- Division of Pediatric Surgery, Michael E. DeBakey Department of Surgery, Baylor College of Medicine, Houston, Texas, United States
| | - Eugene S Kim
- Division of Pediatric Surgery, Michael E. DeBakey Department of Surgery, Baylor College of Medicine, Houston, Texas, United States
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Abstract
Most of the children operated for esophageal atresia will survive the neonatal period. However, medium-term and late complications are frequent in this population. Gastroesophageal reflux disease is observed in 26 to 75% of the cases and can be responsible for peptic esophagitis, anastomotic stenosis, and Barrett esophagus, which is a risk factor for adenocarcinoma of the esophagus. Dysphagia is frequently observed, sometimes several years after the surgery, affecting up to 45% of children at the age of 5 years. Growth retardation is present in nearly one-third of children at the age of 5 years. Ear, nose, and throat and respiratory complications are also very frequent but tend to improve with time. Tracheomalacia is found in 75% of these children at birth, sometimes responsible for severe complications (malaise, bradycardia). Respiratory symptoms are dominated by chronic cough, wheezing, and infections reported in 29% of the children by the age of 5 years. Restrictive, obstructive syndromes and bronchial hyperactivity can be observed, but usually remain moderate. All these complications can influence the patient's quality of life, which is moderately impaired compared to healthy controls. The high frequency of late sequelae in esophageal atresia justifies regular and multidisciplinary follow-up through adulthood.
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