Pediatric benign esophageal strictures: current understanding from etiology to treatment

Introduction

Esophageal strictures (ES) constitute a substantial clinical burden in the pediatric population, manifesting as dysphagia, nutritional compromise, growth faltering, and recurrent hospitalizations (1). Affected children often experience considerable morbidity and may require repeated endoscopic dilation or surgical resection to relieve obstruction. Current classification systems categorize ES into two distinct phenotypes—simple or complex strictures—based on morphological characteristics such as size, symmetry, and endoscopic accessibility. Simple strictures are characterized by short-segment involvement (<2 cm), concentric luminal narrowing, and endoscopic permeability (8–9.8 mm scope passage). In contrast, complex strictures are characterized by long-segment pathology (≥2 cm), tortuous configurations, or complete luminal obliteration (1,2). For the majority of simple benign strictures, minimally invasive endoscopic therapy is beneficial. In contrast, complex strictures are more resistant to initial medical and endoscopic therapies (2). A subset of refractory ES, commonly arising from caustic ingestion, surgical anastomosis, congenital malformations, or radiation-induced strictures, poses particular therapeutic challenges because of the absence of uniform definition of refractory ES (2,3). The mainstream definition includes (meet one of the following): failure to achieve age-adjusted luminal patency after ≥5 dilations within 5 months, or the need for ≥7 dilation procedures irrespective of the time frame, or persistent dysphagia (pediatric eating assessment tool score ≥4) despite 6 months of multimodal therapy (1,3). This comprehensive review aimed to provide an updated overview of pediatric ES management and available evidence, focusing on the most recent technologies, to guide clinicians in effectively managing complex cases.

Etiology

ES is common in children and usually benign in nature (4). The causes can be classified as congenital, secondary, and dyskinetic types (Table 1). ES can also be grouped into three categories: (I) intrinsic conditions that narrow the esophageal lumen through inflammation, fibrosis, or neoplasia; (II) extrinsic conditions that compromise the esophageal lumen by direct invasion or lymph node enlargement; (III) diseases that disrupt esophageal peristalsis or lower esophageal sphincter function by affecting the esophageal smooth muscle and its innervation (5).

The estimated incidence rate of congenital esophageal stenosis (CES) ranges from 1/25,000 to 1/50,000 live births (6,7). It may present subtly, sometimes observed in the neonatal period as recurrent vomiting during feeding, or later in infancy with the introduction of solids (8). The age of presentation can vary considerably and depends on the severity of the stenosis. CES is typically located in the middle and lower part of the esophagus and is classified into the following three subtypes: fibromuscular thickening (54%), tracheobronchial remnants (30%), or membranous web (15%), with approximately 10–14% being associated with esophageal atresia (EA) as a second lesion (4,6,7,9). Endoscopic ultrasonography has shown promise in classifying this disease, while reports on its application in preschool children are scarce (10). In most cases, surgical specimens cannot be procured, making histological classification relatively difficult (7).

Secondary ES are the most common type of ES in children. Major causes include corrosive and button battery ingestion, anastomotic strictures after tracheo-oesophageal fistula/oesophageal atresia (TOF/OA), gastroesophageal reflux disease (GERD), eosinophilic esophagitis (EoE), and epidermolysis bullosa. Less common causes include esophageal ganglioneuromatosis, actinomycosis, scald injury, and radiation-induced injury (3,4,7,11-15). Corrosive ingestion is an important cause of both ES and refractory ES. The amount, physical state, and pH of the corrosives significantly affect the location and severity of esophageal tissue damage (16,17). Upon contact with strong bases and acids, mucosal damage begins immediately. Acid ingestion usually triggers coagulation necrosis, leading to mucosal scarring. In contrast, alkaline substances can penetrate tissues and induce liquefactive necrosis. Tissue damage continues until the alkalis are neutralized (16). By the third week after ingesting corrosive agents, fibroblast proliferation results in the formation of ES and scar tissue within the submucosal and muscular layers (16,18). Accidental ingestion and impaction of button batteries are also relatively common causes of serious esophageal complications. Button batteries can induce liquefactive necrosis at the negative pole. They are most commonly ingested by toddlers aged 1–3 years (19). Caustic injury can occur within 2 h of exposure and may progress even after the battery is removed (19). ES following tracheoesophageal fistula (TEF)/EA repair is the most common postoperative complication, occurring in 40–56% of cases (12,20). Additionally, up to 70% of infants with TEF/EA develop coexistent GERD, which significantly contributes to the pathogenesis of postoperative strictures (21). EoE is a chronic, immune-mediated inflammatory disease of the esophagus. The estimated incidence in children is 6.6/100,000/year, though it varies geographically. Exposure to foods commonly present in the diet is associated with esophageal mucosal infiltration by mixed granulocytes. The inflammation weakens the integrity of the epithelial barrier, damages the mucosa, and is associated with progressive esophageal fibrosis (22). Adolescents and adults have typical symptoms such as solid food dysphagia and esophageal food impaction. However, these symptoms are less specific in younger children and infants (22,23).

Motility-associated ES primarily results from the failure of the lower esophageal sphincter to achieve effective relaxation, often accompanied by impaired esophageal peristalsis, such as achalasia. The estimated incidence is approximately 0.1–0.3 cases per 100,000 children annually (24). The leading pathogenic hypothesis involves a virus-driven autoimmune neurodegenerative process affecting the myenteric plexus that innervates the esophageal smooth muscle.

Diagnosis and assessment

Children with ES typically exhibit similar clinical manifestations despite varying underlying etiologies. Symptom severity often depends on the location, severity, and degree of stenosis. Dysphagia is the main and typical symptom of ES, which involves normal swallowing of liquids but difficulty with semisolid or solid foods (12). In infants and young children, this condition may present as food refusal, excessive salivation, feeding difficulties, gastroesophageal reflux, or malnutrition. Additionally, some patients may exhibit respiratory symptoms such as recurrent coughing, wheezing, or pneumonia (1,12,25). A detailed medical history should be obtained when assessing patients with suspected ES. The essential information includes dietary habits, nutritional status, history of accidental ingestion or swallowing, prior surgeries, and history of radiation therapy.

The condition can be evaluated more precisely using the following examination methods: (I) esophagography and gastroscopy: these can determine the location, range, form, and degree of stenosis or the presence of a pseudodiverticulum; observe the mucosal lesions; or obtain biopsy samples from strictures for analysis to exclude malignancy and EoE (1,26,27). (II) Cervicothoracic computed tomography (CT) or contrast-enhanced CT: CT imaging can help assess the esophageal wall or other compression-associated related strictures, such as vascular rings and slings, derived from malformations of the primitive aortic arch system or tumors (12). (III) Mini-probe endoscopic ultrasound: high-frequency mini-probes can visualize individual layers of the gastrointestinal wall and are suitable for use in the preschool pediatric population, as they can pass through a 2.8-mm channel of a standard endoscope. Mini-probe endoscopic ultrasound can provide detailed information about the esophageal wall in patients with ES, including wall thickness, extent of involvement, and proximity to the surrounding organs, helping to predict the effects of expansion and differentiate tracheobronchial remnants from other CES (1,10,28). (IV) High-resolution esophageal manometry: this is the gold standard for diagnosing esophageal motility disorders, such as achalasia, esophagogastric junction outflow obstruction, and ineffective motility. It can measure the intraluminal pressure along the esophagus, which is then converted it into dynamic esophageal pressure topography plots (29). These plots provide real-time measurements of contractility, sphincter amplitude, and bolus transit, resulting in an overall assessment of esophageal motility using standardized algorithms, such as the Chicago classification system (29).

Treatment

The goal of stricture treatment is to relieve symptoms, permit unrestricted oral diet, and reduce the risk of pulmonary aspiration (30). Management of benign ES requires a dual approach: mechanical dilation to relieve luminal obstruction and targeted therapy to address the underlying etiology. Treatment response depends not only on the stricture’s etiology but also on other factors such as its length and diameter, depth of tissue injury, and presence of comorbidities (4). Treatment timing and strategies should be individualized. We reviewed the relevant studies about the treatment outcomes of pediatric benign ES in recent 10 years (Table 2). The following summarizes the current treatment methods used for ES.

Endoscopic dilations

Endoscopic dilation is the initial choice for most patients, owing to its relatively low risk and positive efficacy. The two common types of dilators used are guidewire-directed bougie dilators and through-the-scope balloon dilators. Balloon dilators apply a uniform radial force across the stricture, whereas bougie exert both radial and longitudinal shear forces (25). Bougie dilators allow the operator to assess the degree of resistance during dilation and help in determining bougie size in subsequent dilations. They can be used for simple strictures and proximal ES, especially anastomotic strictures (2). Balloon dilation allows direct visualization during the procedure and can be performed under fluoroscopic or endoscopic guidance. Fluoroscopic dilation is usually performed by interventional radiologists, while endoscopic dilation is performed by gastroenterologists (25). An absolute contraindication to dilation is esophageal perforation. A retrospective analysis of 60 pediatric patients with ES treated with bougie dilation reported favorable outcomes in Groups A (peptic stricture, 70%), B (caustic stricture, 50%), and C (strictures following EA surgery, 50%). Additionally, 71.6% of strictures were impassable during the first expansion; however, subsequent dilations enabled passage in 86.6% of the impassable strictures (97). He et al. (39) evaluated 75 patients with postoperative anastomotic stenosis following EA, who underwent a total of 210 bougie dilations (median of three dilations per patient). The overall effectiveness was 98.67% (74/75), with complications including perforation in two patients (0.95%) and evident bleeding in three patients (1.43%). In a Turkish study involving 44 ES cases, mostly due to corrosive ingestion or anastomosis (72.2%), 52.6% were treated with Savary-Gilliard bougie while the remaining patients were treated with balloon dilators. The complication rates for the balloon and bougie dilation sessions were 2.4% and 2.1%, respectively (44). Kahriman et al. (52) retrospectively reviewed 375 balloon dilation procedures in 116 patients, reporting a clinical success rate of 85% per patients, with an overall complication rate of 0.5% per procedure and a perforation rate of 0.25%. Another study compared the efficacy and safety of endoscopic and radiologic balloon dilations in ES treatment and revealed that both methods were equally effective; however, radiologic dilations were associated with a significantly higher complication rate (25). Overall, no significant differences in safety and efficacy have been found between bougie and balloon dilators in pediatric patients according to retrospective studies (4,98). Therefore, the choice of dilator is based on the clinician’s preference, local expertise, and equipment availability. Additionally, complex strictures that are longer than 2 cm, irregular, angular, or with a narrowed lumen require multiple dilations and are associated with higher recurrence rates (28). In patients with anastomotic strictures following EA repair, factors such as repeated balloon dilation, older age, and eccentric stricture shape are associated with a poor response to esophageal balloon dilation (47). Predictors of dilation failure requiring stricture resection include long-gap EA, stricture length ≥10 mm, prior anastomotic leakage, and the requirement for ≥7 balloon dilations (1,99). The most frequently reported complications of esophageal dilation are perforation, hemorrhage, and bacteremia.

Both dilation techniques should start with small diameter dilation and progress slowly at each session until the target diameter or resolution of symptoms is achieved, that is, “start small and go slow”. The “rule of three” is routinely applied during each dilation session: dilate maximal up to three times the diameter of stenosis, with an average of three dilations and a minimal period of three weeks between two dilation sessions. Generally, the balloon is 1 to 2 mm larger than the narrowed opening according to the preoperative assessment, and performed no more than three minutes and three dilations in 1 mm increments per session is recommended (23,100). When serial dilations are required, the common practice is to limit the dilation to no more than 3 mm per session (101). Endoscopic dilation is a therapeutic method for managing ES, rings, and narrow-caliber esophagi in EoE. However, dilation cannot improve esophageal inflammation or ongoing mucosal damage, so inflammation should be controlled before initiating dilation. Nonetheless, in cases of medication nonadherence or lack of response to medication, high-grade stenosis, or recurrent food impaction, esophageal dilation may be considered before inflammation control (22,23).

Esophageal stenting

Esophageal stent placement offers potential benefits because of its continuous expansion force, which may lead to stricture remodeling and effectively reduce bleeding and perforation risks. Nitinol fully covered self-expandable metal stents (FCSEMS) are widely used for the endoscopic therapy of ES in adults. However, esophageal stent placement is generally considered a second-line treatment owing to its relatively high rate of adverse events and cost. It is recommended for refractory ES, esophageal fistulas, and perforations (102). In 2021, the guidelines of the European Society of Gastrointestinal Endoscopy were established for the use of esophageal stents in both benign and malignant diseases in adults (102). In contrast, significant heterogeneity exists in pediatric stenting practices owing to the variety of stent types and sizes used, and no clear guidelines currently exist for esophageal stenting in children.

The most common stents include FCSEMS, self-expandable metal stents (SEMS), and silastic esophageal stents, although biodegradable stents (BDS) were reported as well (48). Esophageal stents are ideally used for strictures located in the middle or lower segments of the esophagus. If the stricture is too high, the stent may compress the airway, causing frequent coughing and breathing difficulties, and may also interfere with the upper esophageal sphincter, resulting in frequent vomiting and dysphagia. Conversely, if the stricture is too low, the risks of GERD and aspiration after stent placement are high. A study reported that stent therapy successfully prevented surgical stricture resection at the site of EA repair in 41% of a pediatric cohort (49 patients), with good long-term follow-up (53). The same study also found that when the stricture recoiled to greater than 4 mm between the time of stent removal and subsequent endoscopy, the odds of the stricture being refractory and requiring surgical stricture resection increased (53). A systematic review of 340 pediatric patients across 18 studies revealed that stenting was successful in 54% of the participants, partially successful in 28% (requiring additional dilations or pharmacological therapy), and ineffective in 14% (48). Major complications included stent migration or displacement (45 cases), severe gastroesophageal reflux (18 cases), perforations (15 cases), aortic or subclavian lesions (4 cases), airway disorders (4 cases), fistula (3 cases), pneumonia (2 cases), and 1 case each of mediastinitis, cervical abscess, and deep ulceration (48). Wang et al. (55) reported successful FCSEMS placement in 14 pediatric patients with either perforation or refractory strictures. During the 5 to 83 months’ follow-up, dysphagia was significantly alleviated in 13 patients, with Ogilvie and Atkinson scores improving from grades III–IV to 0–I. The stricture diameter expanded from 2–5 mm pre-treatment to 9–14 mm after stenting. Yen et al. (63) reported successful retrograde esophageal stenting for ES following EA repair in a 3-month-old girl. The case involved a complication after esophageal dilation that may have precluded subsequent endoscopic therapy because the guidewire could not pass through the oral route. Awolaran et al. (64) reported their experience with BDS in three children with severe refractory ES, none of whom showed clinical benefit. Although the advantage of avoiding the need to remove the stent is attractive in pediatrics, BDS have yet to be proven as effective options. Currently, no consensus exists regarding the optimal stent dwell time in pediatrics, and patient’s comfort is a crucial factor affecting the duration of stent placement. Many children experience discomfort during the first 24–72 h after placement, but their tolerance improves over time. Adverse events include stent migration, esophageal ulceration, chest pain, granulation tissue formation, retching, aspiration, breathing difficulty, esophageal perforation, and stent erosion into the vasculature or airway (1). The most common complications are stent migration, granulation tissue formation and stenosis recurrence, which often necessitate stent replacement or change to other methods (48).

Endoscopic incisional therapy (EIT)

EIT shows promise as an adjunct treatment option for pediatric refractory ES and may be considered before surgical resection. EIT involves single or multiple radial cuts of the ES using an electrosurgical knife passed through the working channel of an endoscope. This technique is typically reserved for short strictures, particularly when a hard or fibrotic, rather than soft or inflammatory, “ring” or “shelf” is identified (4). Patients with asymmetric strictures may particularly benefit from EIT (3,4).

According to a study in which 21 of 36 patients with CES underwent EIT, total or partial oral feeding was achieved more frequently in patients treated with EIT than in those who received non-EIT therapeutic endoscopy (81% vs. 38%, respectively). In contrast, the need for surgical intervention was considerably lower in the EIT group (14% vs. 62%). EIT expands the therapeutic options for physicians and may help avoid surgical intervention in CES cases (67). Another study involving 57 patients demonstrated that EIT therapy was successful in 100% of cases with non-refractory anastomotic strictures and 61% of cases with refractory anastomotic strictures. The youngest patient was 3 months old, and the overall adverse event rate was 5.3% (3). The major complication of EIT was esophageal leak or perforation. However, given that the complication rate of EIT is higher than that of standard dilation, it should only be performed by experienced therapeutic endoscopists.

Pharmacological treatment

Systemic or intralesional steroids, including triamcinolone acetonide, dexamethasone, and methylprednisolone, inhibit inflammatory responses, reduce collagen synthesis, and alleviate fibrosis and scar formation. Triamcinolone acetonide or acetate is typically injected into the four quadrants of the esophagus after endoscopic dilation, targeting the scar tissue or mucosal breaks (1). A meta-analysis of three randomized controlled trials involving adults with anastomotic strictures demonstrated that patients in the steroid group (n=72) required significantly fewer dilations than those in the placebo group (n=72) (103). Similarly, a retrospective case series of six children who had experienced multiple endoscopic dilations owing to recurrent anastomotic strictures following EA surgery revealed that intralesional steroid injection (ISI) combined with dilation was effective in five patients (83.3%) who eventually required no further dilations (20). Another study described ISI without dilation as the treatment for impassable caustic ES. ISI was administered to 340 patients during the initial endoscopy, conducted 6 weeks after corrosive ingestion. In 75% of cases, a second endoscopy 2 weeks later enabled successful dilation, suggesting that ISI is a feasible rescue therapy for impassable caustic strictures and may help avoid surgery (70). An international survey on anastomotic stricture management following EA repair analyzed responses from 115 centers across 32 countries. The preferred first-line adjunctive treatments for recurrent strictures were intralesional steroids and topical mitomycin C (MMC), which were used in 47% and 31% of the centers, respectively (30). However, current evidence does not clearly define the number of intralesional steroid sessions recommended per patient or the ideal treatment duration. Potential complications of ISI include adrenal suppression, perforation, intramural infection, candida infection, mediastinitis, and pleural effusion (20). In contrast, swallowed topical steroids, such as fluticasone or budesonide viscous formulations, are highly effective in inducing EoE remission and are preferred over systemic steroids because of their favorable safety profile (104). Nevertheless, a study involving 20 children with varying degrees of ES secondary to EoE reported that systemic steroid therapy led to clinical symptom improvement in all patients, with endoscopic evidence of stenosis regression in 19 cases. Only mild adverse effects were observed, suggesting that patients may benefit from short-term systemic steroids as an alternative to mechanical dilation (105).

Fibroblast proliferation and collagen formation are the crucial steps in the initiation of scar formation following mucosal injury. MMC can inhibit cell division, protein synthesis, and fibroblast proliferation, thereby reducing scar formation (51). A prospective controlled study in children with post-corrosive ES requiring esophageal dilation revealed a significant improvement in dysphagia grade in the group receiving MMC combined with dilation (106). Another study demonstrated that topical application of MMC on long strictures as an adjuvant therapy to endoscopic dilation achieved a success rate of 85.7%, reducing the need for esophageal replacement (107). In a prospective, randomized controlled trial by Sawires et al. (35), 61 children with a history of caustic ingestion were assigned to two groups: Group A (n=30) received topical MMC application within the initial 48 h, while Group B (n=31) received only conventional management. Complete response was achieved in 26 patients (86.7%) in Group A compared to 11 patients (35.5%) in Group B, highlighting the efficacy of early MMC application over conventional endoscopic dilation. The application of MMC was usually initiated 2 weeks after the first dilation session. Topical application through a multi-porous spray catheter placed on the injury under direct visualization. The most commonly used concentration is 0.4 mg/mL. Length of application in the studies varies from 1 to 5 minutes. Despite its therapeutic benefits in ES, MMC poses carcinogenic risks to normal mucosal tissue because of its chemotherapeutic nature. Moreover, reference standards for its optimal frequency of use or total dosage have not been established.

Gastric acid-induced reflux esophagitis causes collagen and fibrous tissue deposition, leading to stricture formation. Persistent acid exposure of the esophagus results in fibrosis, contributing to the development of benign refractory strictures. Therefore, protecting the esophageal mucosa barrier from acid reflux can be achieved through gastric acid suppression. Proton pump inhibitors are the most commonly used acid-suppressing agents. Additionally, sucralfate can provide a mechanical barrier by coating the esophageal mucosa at the site of injury, thereby preventing further injury. In a study by Akhijahani et al (108), 60 children with esophageal burns due to the ingestion of caustic agents were divided into two groups. The incidence of ES was significantly lower in the sucralfate intervention group than in the standard treatment group (37% vs. 67%, P=0.042). However, relevant literature is scarce, and further verification is needed.

Magnetic recanalization

Magnetic recanalization is a novel and effective treatment for children with refractory ES. Although it has been applied in some international medical centers, unified management and standardized clinical procedures are lacking. Indications for magnetic recanalization include: (I) various types of ES (postoperative, congenital, or burn-related) generally <2 cm and 2–3 cm can be attempted; and (II) EA up to 3 cm cannot be attempted. Contraindications include: (I) combined esophagotracheal fistula, (II) combined esophageal perforation, (III) stenosis exceeding 4 cm in length, and (IV) ectopic tracheal cartilage in CES (109). The operation process includes: the upper magnet is placed in the upper esophageal pouch and is attached to an orogastric suction tube that controls secretions, while the lower magnet is advanced into the lower esophageal pouch (usually through a gastrostomy). Then two magnets naturally meet and quickly compresses the scar tissue in the stenosis (110). A study reviewed the use of neodymium-iron-boron magnets for treating refractory strictures in two surgical candidates. Magnets were left in place for 7–10 days to achieve esophageal patency, followed by the placement of self-expanding stents, and both patients achieved durable esophageal patency (86).

Surgical treatment

Surgical treatment of ES includes resection of the stenotic segment, followed by end-to-end anastomosis and esophageal replacement. Resection with anastomosis is applicable to tracheobronchial remnants or short-segment stenosis (≤2 cm) that are unresponsive to repeated endoscopic treatments (57,85,111). Esophageal replacement is considered for long-segment fibromuscular thickening or corrosive ES that is unresponsive to multiple endoscopic treatments (106). Currently, the commonly used esophageal substitution surgeries include colonic interposition, gastric pull-up, gastric tube reconstruction, and small intestine interposition. The main postoperative complications include anastomotic leak or strictures, infection, and secondary reflux (112).

Conclusions

Esophageal dilation is generally regarded as the preferred and most effective treatment method for various ES types, provided there are no contraindications. Various minimally invasive endoscopic techniques have demonstrated promising outcomes in ES treatment and may help pediatric patients avoid surgery. Moreover, many serious complications related to endoscopy have a relatively low incidence rate, and conservative treatment can be attempted to actively save the situation. Surgical intervention is the final choice when other endoscopic techniques have failed or there are severe complications, because it may fail to produce durable response and secondary anastomotic strictures still require ongoing dilation. Based on the literature review and our experience of treating ES, we have proposed an algorithm for the treatment (Figure 1). However, inconsistencies in several crucial definitions, such as refractory or recurrent stenosis, and the criteria for age-appropriate esophageal lumen, have hindered the comparability of existing studies. Currently, no universally accepted consensus exists for the management of refractory or recurrent stenosis. Most existing studies are retrospective and vary in their definition of key concepts, further limiting their comparability. In the future, more prospective controlled studies examining pediatric populations are needed. Moreover, the management of refractory strictures requires a multidisciplinary approach, involving a wide range of specialties, including surgeons, anesthesiologists, gastroenterologists, and radiologists.

Figure 1 Proposed algorithm for treatment management of ES. CES, congenital esophageal stenosis; EIT, endoscopic incisional therapy; ES, esophageal stricture; ISI, intralesional steroid injection; MMC, mitomycin C.

Acknowledgments

None.

Footnote

Peer Review File: Available at https://tp.amegroups.com/article/view/10.21037/tp-2025-540/prf

Funding: This study was funded by the National Key Research and Development Program of China (Nos. 2023YFC2706500 and 2023YFC2706504).

Conflicts of Interest: All authors have completed the ICMJE uniform disclosure form (available at https://tp.amegroups.com/article/view/10.21037/tp-2025-540/coif). The authors have no conflicts of interest to declare.

Ethical Statement: The authors are accountable for all aspects of the work in ensuring that questions related to the accuracy or integrity of any part of the work are appropriately investigated and resolved.

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