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Revisional Surgery for Recurrent Tracheoesophageal Fistula and Anastomotic Complications after Repair of Oesophageal Atresia in 258 Infants
Abstract
Aim:
We assessed the occurrence and outcome of major reoperations following repair of esophageal atresia with or without tracheoesophageal fistula (TOF). Major outcome measures were survival, preservation of native esophagus, and long-term esophageal function.
Methods:
Hospital charts of 258 consecutive patients treated for esophageal atresia from 1980 to 2013 were reviewed.
Results:
Forty-two (16%) patients required a total of 57 reoperations after primary repair (n=37) or esophageal reconstruction (n=5). The indications were anastomotic leakage (n=17), anastomotic rupture after endoscopic dilatation (n=5), recurrent tracheoesophageal fistula (TOF) (n=12), undiagnosed proximal TOF (n=3), recalcitrant anastomotic stricture (n=11, primary anastomosis 9, reconstruction 2), undetected proximal fistula (n=3), and inadvertently perforated jejunal graft (n=1). Anastomotic leakage and rupture after dilatation were treated with rethoracotomy and suture and recurrent or undetected TOF by open repair. Strictures not manageable with repeated dilatations were resected and esophageal ends reanastomosed (n=10) or bridged with jejunum graft (n=1). Five (12%) patients required further reoperations, two after recurrent TEF (reocclusion n=1, reconstruction with gastric tube n=1), two after stricture operations (re-resection n=1, resuture after leakage n=1), and one after recurrent dilatation-related rupture. Mortality was 4/42 (10%). Two patients died of recurred leakage or TOF and two of unrelated cause. Of 38 survivors, 35 retained their native or initially reconstructed esophagus, and 3 had secondary reconstruction. After a median follow-up of 23 (range 0.6-32) years, 35 (95%) patients have acceptable esophageal function. Three patients remained dependent on gastrostomy feedings.
Conclusion:
Anastomotic and TOF complications required a substantial number of reoperations, including esophageal reconstructions. Over 90% of the patients survived with a functioning native or reconstructed esophagus.
... One of the most common reasons for reoperation after esophageal atresia repair is undiagnosed upper tracheoesophageal fistula or recurrent tracheoesophageal fistulas (RTEF), occurring in 1.9-16% of cases [1][2][3][4][5]. Although RTEF is rare, it is also one of the most complex and serious late complications of esophageal atresia surgery. ...
... Not only fistulas but also tracheal diverticula (TD) as a remnant of the closed fistula may be seen. RTEF is usually a consequence of complications, mainly leakage, after primary right-sided repair of esophageal atresia [2][3][4]. It results in extensive adhesions and scarring developing in the surrounding tissues and pleural cavity. ...
... Current treatment is endoscopic obliteration of the fistula or open right-sided surgical removal of the fistula or diverticulum. The reoperation is either open or endoscopic, the recanalization rate is still over 20%, and the mortality is up to 10% [2][3][4][6][7][8][9]. ...
Background: Recurrent tracheoesophageal fistula (RTEF) is usually a consequence of leakage or other complications after esophageal atresia repair performed through right-sided access. This results in extensive intrapleural adhesions, and open redo surgery poses a challenge. Alternatively, endoscopic endotracheal fistula obliteration usually requires repetitive procedures, and its success rate varies significantly between centers. We present a novel approach to recurrent fistulas. The innovation is in reaching the fistula through the virgin field via left-sided three-port thoracoscopy instead of classical right-sided thoracotomy.
Methods: This is a presentation of a new operative technique based on a retrospective case series of patients operated on at our department between 2016 and 2023.
Results: Eight patients after esophageal atresia repair (six with RTEF and two with post-fistula tracheal diverticula) were successfully treated with left-sided thoracoscopy. There were no conversions. One patient required rethoracoscopy for chylothorax. Another one, after RTEF closure, underwent multiple endoscopic obliterations of subsequent tracheal diverticulum. No other major complications nor re-recurrences were noted.
Conclusions: Left-sided thoracoscopy in redo esophageal atresia has the advantage of a “virgin” operative field and grants feasible access to the RTEF or tracheal diverticulum. We believe that this approach is worth further exploration because it combines minimal invasiveness with high effectiveness without all the consequences of a thoracotomy.
... Conversely, major anastomotic leaks should instead be operated on while the anastomotic edge is still fresh for re-suturing before inflammation and necrosis can cause tissue damage. The rationale for the intervention is to prevent serious and potentially fatal complications, such as mediastinitis, mediastinal abscess, sepsis, and tension pneumothorax (1,11,22). ...
... Moreover, any vascular compromise during the mobilization of the lower esophagus which has a segmental blood supply from the aorta and the intercostal vessels, may yield to ischemia at the esophageal anastomosis, which may lead to stricture formation (7). Patients with long gap EA undergoing a staged repair have been reported to be at higher risk of stricture formation over those who undergo primary repair (22,27). Nonetheless, the Midwest Pediatric Surgery Consortium reported no differences in stricture rate between patients with and without a long gap EA (3). ...
... Nonetheless, the Midwest Pediatric Surgery Consortium reported no differences in stricture rate between patients with and without a long gap EA (3). Several studies have shown that the risk of esophageal stricture is significantly higher in infants that develop an anastomotic leak compared to those without leak, possibly due to local inflammation and scar formation (11,13,22). GER has always been considered a significant predisposing factor for stricture formation (15,22,28,29) and recent evidence shows that a third of EA/TEF patients with clinico-pathological evidence of eosinophilic esophagitis had a history of recurrent stricture (30). ...
... The standard approach to the repair of EA and TEF is well described which consists of ligation of fistula and primary anastomosis of esophagus. Although EA repair has usually excellent outcome, several post-operative challenging situations may come through including fail of repair, need for lengthening procedures, need for revisional surgeries, and several defined complications (4,7). Among those, the fistula occurrence after primary repair of EA is one of the most challenging complications (4). ...
... The diagnosis of recTEF is also challenging. The patients with EA very commonly experience several respiratory symptoms such as coughing, chocking or recurrent respiratory tract infections (1,6,7,11). Since all of the patients either with gastroesophageal reflux, tracheomalacia or recTEF experience similar symptoms, it is difficult to distinguish which of the EA-related complications is the cause of these symptoms. ...
... The diagnostic modalities include esophagogram and bronchoscopy (6,7,11). The routine esophagogram may miss recTEF very commonly. ...
... We present a child that had 3 TEF recurrences which were all appropriately managed with commonly used surgical techniques. [1][2][3] Below we report on the technique we used in the last repair where an omental flap was placed between the esophageal anastomosis and tracheal suture line. ...
... Our patient's first recurrent TEF repair required esophageal resection due to concurrent stricture and a new anastomosis which has been associated with a higher likelihood of complications. 2 With the exception of the last repair, each procedure was complicated by an esophageal leak, which is a significant risk factor for stricture (relative risk [RR] = 25, 95% CI: 5.8-111, P < .001) and recurrent TEF (RR = 8.0, 95% CI: 2.1-3.1, ...
... P = .001). 2 As the esophageal contents leak out, it forms a localized abscess that penetrates into the trachea creating a fistula tract. Additionally, our patient struggled with reflux, aspiration, and multiple respiratory infections which increased inflammation in the thorax and are known contributors to the pathology of recurrent TEF. 1 During our patient's second recurrent TEF repair, it was noted that old suture material was present in the trachea which may have led to a foreign body reaction and recurrent fistula formation. ...
A term female infant with tracheoesophageal fistula (TEF) and esophageal atresia (EA) underwent primary operative repair that failed with 3 TEF recurrences, which all presented with feeding and respiratory issues. Recurrences were managed with reoperation and an interpositional flap of pleura and a flap of intercostal muscle on 2 separate occasions. The third recurrence was managed with complete dissection of the esophagus prior to the division of the fistula and the interposition of an omental flap between the esophageal and tracheal repair. We present the use of a viable omental flap and complete esophageal mobilization to prevent subsequent TEF recurrences and avoid the additional morbidity of reconstructive surgery.
... Recurrent tracheoesophageal fistula (TEF) is a common complication of congenital esophageal atresia repair, occurring in up to 5-10% of patients [1][2][3], and is more likely to be associated with anastomotic leakage [2,3]. Recurrence typically occurs in the neonatal period or during early infancy [2,4], and late recurrence in the adult period is very rare. ...
... Recurrent tracheoesophageal fistula (TEF) is a common complication of congenital esophageal atresia repair, occurring in up to 5-10% of patients [1][2][3], and is more likely to be associated with anastomotic leakage [2,3]. Recurrence typically occurs in the neonatal period or during early infancy [2,4], and late recurrence in the adult period is very rare. ...
... Reoperation for recurrent TEF is often complex and still remains challenging because of various complications, such as leakage, stricture, re-recurrent TEF, and mortality [2][3][4]. Interposition of a natural flap is one of the most essential steps during reoperation. Autologous grafts traditionally include pleura, intercostal muscle, and pericardium [2,5], although the ideal graft for interposition in TEF repair is still debated. ...
... 9 Several studies have found that preoperative diagnosis of type D is difficult, and most cases are misdiagnosed as type C before surgery. 9,17,18 Only two of the 14 cases in this study were diagnosed preoperatively. Seven patients were diagnosed with type D during the initial surgery. ...
... 11,21 Although studies have suggested that some factors limit routine preoperative bronchoscopy in the neonatal period, including low incidence of D-type EA/TEF, poor tolerance in neonates, and relatively hidden proximal fistula requiring experienced endoscopists. 9,17,22 We still recommend that routine preoperative bronchoscopy be considered in the neonatal period if the facility has adequate medical resources. Especially, in patients with a narrow proximal blind-ending pouch on esophagography, or when the surgeon lacks sufficient experience. ...
Importance
Type D esophageal atresia (EA) with tracheoesophageal fistula (TEF) is characterized by EA with both proximal and distal TEFs. It is a rare congenital anomaly with a very low incidence.
Objective
To investigate diagnostic and treatment strategies for this rare condition.
Methods
We retrospectively reviewed the clinicopathological features of patients with EA/TEF treated at our institution between January 2007 and September 2021.
Results
Among 386 patients with EA/TEF, 14 (3.6%) had type D EA/TEF. Only two patients were diagnosed with proximal TEF preoperatively. Seven patients were diagnosed intraoperatively. Five patients were missed for diagnosis during the initial surgery but was later confirmed by bronchoscopy. During the neonatal period, seven patients underwent a one‐stage repair of proximal and distal TEF via thoracoscopy or thoracotomy. Due to missed diagnosis and other reasons, the other 7 patients underwent two‐stage surgery for repair of the proximal TEF, including cervical incision and thoracoscopy. Ten of the 14 patients experienced postoperative complications including anastomotic leakage, pneumothorax, esophageal stricture, and recurrence. Patients who underwent one‐stage repair of distal and proximal TEF during the neonatal period showed a higher incidence of anastomotic leak (4/7). In contrast, only one of seven patients with two‐stage repair of the proximal TEF developed an anastomotic leak.
Interpretation
Type D EA/TEF is a rare condition, and proximal TEFs are easily missed. Bronchoscopy may aim to diagnose and determine the correct surgical approach. A cervical approach may be more suitable for repairing the proximal TEF.
... These fistulas include communications between the esophageal anastomosis and the trachea, a segmental bronchus, pleural cavity or directly pulmonary parenchyma ( Figure 5). Anastomose leak after EA repair is thought to be the leading cause of recurrent TEF (32). Recurrent TEF following anastomose leak due to initial EA repair was reported to be 45.8% to 68% in different studies (18,32,33). ...
... Anastomose leak after EA repair is thought to be the leading cause of recurrent TEF (32). Recurrent TEF following anastomose leak due to initial EA repair was reported to be 45.8% to 68% in different studies (18,32,33). It is also more often seen in children with concomitant congenital esophageal stenosis (14). ...
... Despite the difficulties in the management of refractory and recurrent ASs, the number of reported patients who require resection of the stricture is remarkably small, ranging from 3 to 7% (12,112). No large data are available about long-term outcomes of these patients, since cohorts are small and data are always retrospective. ...
... Resection and esophageal anastomosis is the most common surgical intervention for refractory ASs (112,113). Although mediastinal scarring complicates reoperation, by this time the esophageal ends are in apposition and better vascularized, increasing the likelihood of success (114). ...
... The described tracheomalacia, residual tracheal diverticulum and recurrent TEF have surely contributed to the lower respiratory exacerbations as reported by other studies [42][43][44]. ...
... Because of only few case series describe the presence of residual tracheal diverticulum in children underwent to correction of EA and TEF [42,43], it's not possible to estimate the real frequency of this complication. Only the frequency of recurrent TEF in our study population is higher than previously described [44] and also this finding is greater in children surgically corrected at other centers: this observation is probably influenced by the same selection bias previously discussed for children undergoing Nissen fundoplicatio. ...
Background
Children with congenital esophageal atresia (EA) and tracheoesophageal fistula (TEF) have chronic respiratory symptoms including recurrent pneumonia, wheezing and persistent cough. The aim of this study is to describe the clinical findings of a large group of children with EA and TEF surgically corrected and the instrumental investigation to which they have undergone in order to better understand the patient’s needs and harmonize the care.
Methods
A retrospective data collection was performed on 105 children with EA and TEF followed at Department of Pediatric Medicine of Bambino Gesù Children’s Hospital (Rome, Italy) between 2010 and 2015.
Results
69/105 (66%) children reported lower respiratory symptoms with a mean age onset of 2.2 ± 2.5 years and only 63/69 (91%) performed specialist assessment at Respiratory Unit. Recurrent pneumonia (33%) and wheezing (31%) were the most reported symptoms. The first respiratory evaluation was performed after surgically correction of gastroesophageal reflux (GER) at mean age of 3.9 ± 4.2 years. Twenty nine patients have undergone to chest CT with contrast enhancement detecting localized atelectasis (41%), residual tracheal diverticulum (34%), bronchiectasis (31%), tracheal vascular compression (21%), tracheomalacia (17%) and esophageal diverticulum (14%). Fifty three patients have undergone to airways endoscopy detecting tracheomalacia (66%), residual tracheal diverticulum (26%), recurrent tracheoesophageal fistula (19%) and vocal cord paralysis (11%).
Conclusions
Our study confirms that respiratory symptoms often complicate EA and TEF; their persistence despite medical and surgical treatment of GER means that other etiological hypothesis must be examined and that a complete respiratory diagnostic work up must be considered.
... Despite the difficulties in the management of refractory and recurrent ASs, the number of reported patients who require resection of the stricture is remarkably small, ranging from 3 to 7% (12,112). No large data are available about long-term outcomes of these patients, since cohorts are small and data are always retrospective. ...
... Resection and esophageal anastomosis is the most common surgical intervention for refractory ASs (112,113). Although mediastinal scarring complicates reoperation, by this time the esophageal ends are in apposition and better vascularized, increasing the likelihood of success (114). ...
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.
... In a study by Kamran et al. [76] involving 100 EA patients with refractory AS undergoing surgical stricture repair, it was reported that surgical intervention for refractory AS is associated with low morbidity and a high rate of esophageal preservation. Furthermore, multiple studies have identified severe AS as a significant indication for reoperation following EA repair, with reoperative procedures demonstrating favorable efficacy in managing this complication [77,78]. However, it should be acknowledged that patients undergoing secondary end-to-end anastomosis may still require postoperative dilation, and in certain cases, further revision surgeries may become necessary [75]. ...
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.
... These strictures lead to issues such as growth and developmental limitations due to dietary restrictions, which can be resolved using various nonsurgical treatments such as balloon dilatation, bougienation, and steroid injection [5][6][7] . However, stricture resection and repeat anastomosis are performed when these methods are ineffective 8,9 . ...
Anastomotic stricture is a typical complication of esophageal atresia surgery. Remote ischemic conditioning (RIC) has demonstrated multiorgan benefits, however, its efficacy in the esophagus remains unclear. This study aimed to investigate whether applying RIC after esophageal resection and anastomosis in rats could attenuate esophageal stricture and improve inflammation. Sixty-five male Sprague–Dawley rats were categorized into the following groups: controls with no surgery, resection and anastomosis only, resection and anastomosis with RIC once, and resection and anastomosis with RIC twice. RIC included three cycles of hind-limb ischemia followed by reperfusion. Inflammatory markers associated with the interleukin 6/Janus kinase/ signal transducer and activator of transcription 3 (IL-6/JAK/STAT3) and tumor necrosis factor-alpha/nuclear factor-κB (TNF-α/NF-kB) signaling pathways were evaluated with RNA and protein works. The RIC groups showed significantly lower stricture rates, lower inflammatory markers levels than the resection and anastomosis-only group. The RIC groups had significantly lower IL-6 and TNFa levels than the resection and anastomosis-only group, confirming the inhibitory role of remote ischemic conditioning in the IL-6/JAK/STAT3 and TNF-α/NF-kB signaling pathways. RIC after esophageal resection and anastomosis can reduce the inflammatory response, improving strictures at the esophageal anastomosis site, to be a novel noninvasive intervention for reducing esophageal anastomotic strictures.
... When it was clear that the aforementioned measures had been exhausted without improvement, only then would a child be sent to a pediatric respiratory expert. Several studies have linked tracheomalacia, residual tracheal diverticulum, and recurrent TEF to the worsening of respiratory symptoms in the lower respiratory tract (42)(43)(44)(45). The three factors have worked together to get this result. ...
... Yasuda et al. performed a univariate regression analysis on 177 cases of EA and reported that fundoplication is a significant predictor of the need for EAS resection [16]; however, this predictor did not retain its significance in a subsequent multivariate analysis. If an EAS remains refractory after fundoplication, other treatment plans must be considered [17], including mitomycin C treatment [4,18], endoscopic incisional therapy [19][20][21], and EAS resection with esophageal anastomosis [22]. Researchers have also recommended considering eosinophilic esophagitis in cases of a refractory EAS. ...
Background
An esophageal anastomotic stricture (EAS) after an esophageal atresia surgery occurs in approximately 4–60% of the cases, and its first-line therapy includes balloon dilatation. Oral balloon dilatation cannot be performed in some EAS cases; conversely, even if dilatation is possible, these strictures recur in some cases, necessitating a surgical procedure for repairing the stenosis. However, these procedures are invasive and have short- and long-term complications. If an EAS recurs repeatedly after multiple balloon dilations, gastroesophageal reflux disease (GERD) may be the underlying cause. A fundoplication procedure may be effective for treating a refractory EAS, as in the present case.
Case presentation
A neonatal patient with type D esophageal atresia underwent thoracoscopic esophago-esophageal anastomosis at the age of 1 day, and her postoperative course was uneventful. Thereafter, the patient underwent gastrostomy for poor oral intake at the age of 3 months. After gastrostomy, the patient presented with a complete obstructive EAS. Balloon dilatation via the oral route was attempted; however, a guidewire could not be inserted into the EAS site. Hence, retrograde balloon dilatation via gastrostomy was performed successfully. However, the EAS recurred easily thereafter, and laparoscopic anti-reflux surgery was performed to prevent GERD. The anti-reflux surgery cured the otherwise refractory EAS and prevented its recurrence.
Conclusions
Retrograde balloon dilatation is another treatment option for an EAS. When an EAS recurs soon after dilatation, the patient must be evaluated for GERD; if severe GERD is observed, an appropriate anti-reflux surgery is required before dilating the EAS.
... Reoperation after the repair of esophageal atresia is often complex and requires experienced surgeons. The common condition requiring surgical repair include recurrent tracheoesophageal fistula (rTEF), which occurs in up to 5-14% of patients [1,2], and delayed primary closure. The reoperations of rTEF are mainly divided into endoscopic and operative repairs, and surgical treatment remains dominant [3][4][5]. ...
Revision surgery for the complications after repair of esophageal atresia is often complex because of previous surgeries and chest infections and thus requires surgical expertise. This study describes surgical experiences with the use of indocyanine green (ICG) fluorescence imaging localization-assisted thoracoscopy during revision surgery, including recurrent tracheoesophageal fistula (rTEF) (8 cases, one of which was esophageal-pulmonary fistula) and delayed esophageal closure (1 case). We performed fistula repair and esophageal reconstruction according to the indications of ICG. The application of this method avoids the excessive trauma caused by freeing the trachea and esophagus. Contrast imaging taken one week and one month after surgery indicated no spillover of the contrast agent from the esophagus, except in 1 case. Indocyanine green fluorescence imaging localization-assisted thoracoscopy is worth promoting for revision surgery after esophageal atresia repair.
... Its prevalence in the first year varies from 13% to 57% of patients who have undergone surgical repair. 2 Even with improvement over the years in the general management of patients with OA, anastomotic stricture still complicates the outcomes of patients, with no apparent improvement over time. [3][4][5] Clinically significant anastomotic stricture has been recently defined, in the European Society for Paediatric Gastroenterology, Hepatology and Nutrition/North American Society for Pediatric Gastroenterology, Hepatology and Nutrition consensus on OA, as a narrowing at the level of the anastomosis, diagnosed by contrast and/or endoscopy, that leads to significant functional impairment and associated symptoms. 6 Several predisposing factors have been implicated in anastomotic stricture pathogenesis: long-gap OA, associated tracheomalacia, birth weight, prematurity, gastro-oesophageal reflux, suture material used for anastomosis, anastomosis under tension, method of repair, thoracoscopic surgery and anastomotic leakage. ...
Objectives
To identify the risk factors for anastomotic, refractory and recurrent strictures and to establish whether anastomotic stricture is associated with antireflux surgery.
Design
This prospective national multicentre study included all infants born with oesophageal atresia (OA) over an 8-year period. Data on OA and complications were collected at birth and at 1 year old. Univariate and multivariate analyses were conducted.
Results
1082 patients from 37 centres were included in the study. The prevalence of anastomotic stricture at 1 year old was 23.2%. Anastomosis under tension (defined by the surgeon at the time of repair) and delayed anastomosis (defined as anastomosis performed more than 15 days after birth, excluding delays due to prematurity or severe cardiac diseases) were found to be independent risk factors for anastomotic stricture (2.3 (1.42–3.74) and 4.02 (2.12–7.63), respectively). Patients with anastomotic stricture had a 2.3-fold higher rate of fundoplication compared with others (p=0.001). Anastomosis under tension and delayed anastomosis were found to be independent risk factors for recurrent stricture (1.92 (1.10–3.34) and 5.73 (2.71–12.14), respectively), while delayed anastomosis was the only risk factor for refractory stricture (8.30 (3.34–20.64)). There was a 2.39-fold (1.42–4.04) higher rate of fundoplication in the anastomotic stricture group than in the group without anastomotic stricture (p=0.001).
Conclusions
Patient-related anatomical factors leading to anastomosis under tension and delayed anastomosis increase the risk of anastomotic stricture.
... The main factors that complicate recovery after repair of esophageal atresia (EA) include anastomotic leakage, gastroesophageal reflux (GER), tracheomalacia, recurred tracheoesophageal fistula (TEF), and stenosis of the esophageal anastomosis. 1,2 The obvious symptom of anastomotic stenosis is dysphagia. Anastomotic dysfunction can also cause food and saliva retention in the upper esophagus proximal to the anastomosis and the resulting compression of the trachea by the dilated upper esophagus exacerbates the symptoms of tracheomalacia. ...
Aim To assess the risk factors for anastomotic stricture (AS) in end-to-end anastomosis (EEA) in patients with esophageal atresia (EA).
Methods With ethical consent, hospital records of 341 EA patients from 1980 to 2020 were reviewed. Patients with less than 3 months survival (n = 30) with Gross type E EA (n = 24) and with primary reconstruction (n = 21) were excluded. Outcome measures were revisional surgery for anastomotic stricture (RSAS) and number of dilatations required for anastomotic patency without RSAS. The factors that were tested for risk of RSAS or dilatations were distal tracheoesophageal fistula (TEF) at the carina in C-type EA (congenital TEF [CTEF]), type A/B EA, antireflux surgery (ARS), anastomotic leakage, recurrent TEF, and Spitz group and congenital heart disease.
Main Results A total of 266 patients, Gross type A (n = 17), B (n = 3), C (n = 237), or D (n = 9) underwent EEA (early n = 240, delayed n = 26). Early anastomotic breakdown required secondary reconstruction in five patients. Of the remaining 261 patients, 17 (6.1%) had RSAS, whereas 244 patients with intact end to end required a median of five (interquartile range: 2–8) dilatations for anastomotic patency. Main risk factors for RSAS or (> 8) dilatations were CTEF, type A/B, ARS, and anastomotic leakage that increased the risk of RSAS or dilatations from 4.6- to 11-fold.
Conclusion The risk of severe AS is associated with long-gap EA, significant gastroesophageal reflux, and anastomotic leakage.
... Almost half of our patients did not require dilatation of an anastomotic stricture, whereas stricture formation is a very likely complication following elongation procedures; some authors even report the need for multiple dilatations in all patients following the Fokker procedure (36,(38)(39)(40)(41). We suggest that forming a tension-free anastomosis of two well-perfused esophageal ends, which reduces the risk of a leak, along with a low prevalence of GERD, contributes to our low rate of esophageal strictures; all of these factors are notorious for promoting strictures (42,43). During the 3-month period until the esophagus has grown in length and consists of more resilient tissue is used actively by sham-feeding the patients. ...
The ideal approach to long gap esophageal atresia is still controversial. On one hand, preserving a patient's native esophagus may require several steps and can be fraught with complications. On the other hand, most replacement procedures are irreversible and disrupt gastrointestinal physiology. The purpose of this study was to evaluate the short- and medium-term outcome of electively delayed esophageal elongation procedures before esophageal reconstruction in patients with long-gap esophageal atresia. Since the neonatal esophagus grows over-proportionally and can increase its wall thickness in the first few months of life, we hypothesized that postponing the elongation steps until 3 months of age would lead to a lower complication rate. We thus retrospectively recorded complications such as mediastinitis, anastomotic leakage, stricture formation, or gastroesophageal reflux requiring surgery, and compared it to reported outcomes. In our treatment protocol, patients born with long-gap esophageal atresia underwent gastrostomy placement and were sham fed until 3 months of age. We then assessed the gap between the esophageal ends and started serial elongation procedures. We only proceeded to the reconstruction of the esophagus when its length allowed a tension-free anastomosis. From April 2013 to April 2019, we treated 13 Patients with long-gap esophageal atresia. Nine patients without prior surgical procedures underwent Foker procedures. Four patients arrived with a pre-existing cervical esophagostomy and thus underwent Kimura's procedure, two of them with a concomitant Foker elongation of the lower pouch. Esophageal reconstruction was feasible in all patients, while none of them developed mediastinitis at any point in their treatment. We managed the only anastomotic leak conservatively. Almost half of the patients did not require any further intervention following reconstruction, while three patients required multiple (≥5) anastomotic dilatations. All but one patient achieved full oral nutrition. Only one child required a fundoplication to manage gastroesophageal reflux symptoms. Electively delayed esophageal elongation procedures in patients with long-gap esophageal atresia allowed preservation of the native esophagus in all patients. The approach had low peri-procedural morbidity, and patients enjoy favorable functional outcomes. Therefore, we suggest considering this method in the management of patients with long-gap esophageal atresia.
... Postoperative recurrence of tracheoesophageal fistula (TEF), which arises in 2-11% of patients, is a frequent complication in the repair of esophageal atresia (EA) [1][2][3][4][5][6]. Reoperation against recurrent TEF is usually done with the interposition of the surrounding soft tissues such as the pleura, pericardium, and muscle flaps [2,7]. ...
Background:
Postoperative recurrence of tracheoesophageal fistula (TEF) is a frequent complication in the repair of esophageal atresia (EA). Based on the recent etiologic classification, a TEF that develops in a different new pathway from the original one is categorized as an acquired TEF. The TEFs that fall into this category have been reported to be refractory and their mechanisms have not been fully understood. Here, we report the complicated case of an acquired TEF derived from mediastinitis after the original TEF repair developed an anastomotic stricture. The TEF contained double fistulas, both towards the right lobe bronchi, and was repaired by gastric transposition through a retrosternal route.
Case presentation:
The patient was diagnosed with Gross C esophageal atresia after birth and underwent tracheoesophageal fistula banding during the neonatal period. He experienced an intractable anastomotic stenosis after surgery which was treated with repeated balloon dilation therapy. By the age of 11 months, he developed a mediastinal abscess that improved with conservative treatment. At 18 months old, a fistula from the esophagus to the right superior lobe bronchus was identified. The patient underwent a right upper lobectomy to resect the fistula. However, at 21 months old, another fistula to the right lower lobe was revealed. An esophageal banding was done to relieve the respiratory symptoms. This was followed by esophagectomy and gastric transposition through the retrosternal route at 26 months old. The patient started rehabilitation and oral intake gradually after surgery. By 3 years after gastric transposition, he could already take blended food orally with the support of small amounts of enteral feeding.
Conclusion:
Cases of TEF derived from severe inflammation have the potential to form a complicated network and lead to recurrence. Surgeons should consider the possibility of multiple tiny fistulas in cases of severe acquired TEF. These may be repaired successfully by gastric transposition through the retrosternal route.
... Some authors recommend early thoracotomy and re-anastomosis to optimize outcomes, whereas other authors favored gastric pull-up or colonic or jejunal transposition. [14][15][16] Koivusalo et al. [17] re-operated on 17 out of the 20 patients who developed leakages; anastomotic strictures developed in four patients, and one patient died. Chittmittrapap et al. [10] reported that esophageal leakage developed in 34 (17%) patients and six out of seven undergoing re-operation lost the native esophagus due to major disruptions. ...
Background:
In this study, we aimed to present the results of patients treated for esophageal leakage with a different conservative approach.
Methods:
Ninety-eight patients with esophageal atresia and tracheoesophageal fistula (EA) who underwent surgery in our clinic between February 2013 and January 2018 were retrospectively reviewed in this study. Patients' anastomosis leakage, gestational week, gender, body weight, referral date, recovery time and stenosis were recorded. After leakage detection, the nasogastric catheter was fluoroscopically converted into a nasojejunal catheter using a guidewire and feeding continued.
Results:
Anastomotic leakage developed in 18 (18.3%) patients. The average gestational age at birth was 35.4 weeks; the patients included ten girls and eight boys of average weight 2.41 kg; the average referral period was 2.1 days after birth and the average time of surgery was 2.4 days after birth. The average recovery time was 21.1 days (range: 8-60 days). Eight patients developed stenosis that recovered with dilatation.
Conclusion:
Our findings suggest that our conservative treatment approach, which uses a nasojejunal catheter, is an effective method that would reduce complications, enable earlier feeding, and reduce the cost compared to other treatment approaches.
... This technique can effectively reduce the cost of treatment for the family (P<0.05). Studies have shown that the risk of esophageal stricture in children with anastomotic fistulas is significantly higher than that in children without anastomotic fistulas due to continuous leakage, increasing inflammatory reactions, and scar formation at the anastomotic site [15,16]. Thoracic lavage can drain clean fluid in time and can reduce leakage accumulation at the site of the anastomotic fistula, which can effectively shorten the duration of the anastomotic fistula and reduce the inflammatory reaction at the anastomotic site. ...
Background
The aim of this study was to define whether the addition of thoracic lavage to chest drainage was more efficient than the use of chest drainage alone in the treatment for anastomotic leaks in type III esophageal atresia.
Material/Methods
The clinical data of 42 patients with anastomotic leakage treated with the addition of thoracic lavage from January 2012 to March 2019 in our hospital were analyzed retrospectively. The clinical data of 50 patients with anastomotic leakage treated without thoracic lavage from March 1999 to December 2011 in our hospital were selected as controls.
Results
The duration of fistula healing, mechanical ventilation, hospitalization in intensive care unit, and gastric tube intubation in the thoracic lavage group were significantly shorter than those in the non-lavage group. The cost of hospitalization and the incidence of severe pneumonia were significantly lower in the thoracic lavage group than in the non-lavage group. The diameter of the anastomotic opening after anastomotic fistula healing was wider in the thoracic lavage group than in the non-lavage group.
Conclusions
The technique of thoracic lavage is simple, economical and convenient and can effectively promote the healing of anastomotic fistulas, accelerate postoperative recovery in children and reduce the cost of treatment.
... TOF can be revealed during bronchoscopic exploration either by injection of methylene blue or by the passage of a guidewire through the fistula. 5 Once the diagnosis is made, closure of TOF is indicated. ...
Recurrent tracheo-oesophageal fistula (TOF) is a common complication in children who underwent oesophageal atresia repair. The traditional surgical approach performed either by thoracotomy or cervicotomy is associated with a high rate of morbidity, mortality and new recurrence. In the last decades, endoscopic techniques have emerged as the minimally invasive alternative. However, it seems that the optimal treatment is still unknown. We present a patient with a recurrent TOF who underwent thoracoscopic closure using a 5.8 mm endostapler. The patient was extubated at the end of the procedure, and he started feeding the day after surgery. At 15 months of follow-up, he is asymptomatic. Thoracoscopic closure of TOF using endostaplers seems to be a safe alternative with some possible benefits compared with traditional and endoscopic approach.
... In the case of esophageal atresia and stricture following repair of complicated TEF, minimally invasive techniques may be employed to avoid the morbidity of reoperation. In some studies describing long-term outcomes for TEF, up to 16% of patients required reoperation with an associated mortality of 10% in these patients [3,25]. While an average of 3.2 (range 1-8) esophageal dilations with bougie or pneumatic balloon are effective in the majority of anastomotic strictures, data concerning prognostic factors to predict the trend of dilations and need for further intervention are lacking [26,27]. ...
Anastomotic stricture is a common complication following repair of esophageal atresia (EA). Many factors are thought to contribute to stricture formation and a variety of management techniques have been developed. In this case report, we describe the treatment of a recurrent anastomotic stricture following repair of long-gap esophageal atresia. Porcine bladder extracellular matrix (ECM) was mounted on a stent and delivered endoscopically to the site of recurrent stricture. An appropriate positioning was confirmed using direct endoscopic visualization and intra-operative fluoroscopy. The patient recovered well with persistent radiographic and functional improvements in previous stricture.
Background
Esophageal atresia (EA) with or without tracheoesophageal fistula (TEF) represents a spectrum of relatively rare and complex congenital malformations in newborns. It carries a major challenge for the pediatric surgeon in terms of operative intervention and early identification and management of postoperative complications. The short-and long-term postoperative complications are frequently observed during the follow-up period. This study evaluated the results of 14 cases of EA-TEF to determine the outcome of operative repair.
Methods
This retrospective cohort study included all newborns with EA-TEF who were operated on and followed up in two institutions, King Abdulaziz Medical City (KAMC) and King Faisal Hospital Research Center (KFSHRC) in Jeddah, during a 5-year period (2015–2019). Data include the patient’s age, gender, birth weight, degree of prematurity, type of tracheoesophageal anomaly, the presence of comorbidities, time of intervention, type of operative intervention, and postoperative complications observed during the follow-up period.
Results
The participants included in this study were newborns delivered and operated on at KAMC and KFSHRC. The birth weight was normal >2500 g (64.3%), low birth weight (LBW 1500–2500 g) (28.6%), and extreme low birth weight <1000 g (7.1%). The variants of tracheoesophageal anomalies were Type C (42.9%), Type A (28.6%), and Type E (28.6%). Females were more than males and comorbidity was found in 7 cases. Depending on the type of EA-TEF, the timing of the intervention ranged from 2 to 240 days. Operative interventions were primary anastomosis (35.7%), staged procedure (35.7%), and fistula ligation of Type E (28.6%). Postoperative complications were stricture (50%), gastroesophageal reflux disease (GERD) (28.6%), recurrence (7.1%), tracheomalacia (7.1%), and leak (7.1%). One patient died. All patients with Type C underwent primary anastomosis, except one who underwent a staged operation along with Type A patients. Fistula ligation was achieved in four patients with Type E. The outcomes of patients who received primary anastomosis were stricture (60%), GERD (40%), and recurrence (20%). The patients who underwent staged operation developed strictures (80%), followed by GERD (40.0%), they underwent further fundoplication procedure (40%), tracheomalacia (20%), and leak (20%). GERD developed in 1 of 7 cases (14.3%) who did not have stricture and in 3 of 7 cases (42.9%) who had stricture.
Conclusions
EA/TEF is a rare condition associated with various comorbidities. Postoperative complications include stricture, GERD, recurrence, tracheomalacia, and leak. Further studies analyzing and describing postoperative management in detail are required.
A BSTRACT
Introduction
Anastomotic narrowing post-Type 3 esophageal atresia (EA) primary repair has been linked to life-threatening respiratory complications and failure to thrive during infancy. It becomes important to find some method to ensure anastomotic adequacy in these patients. We in the study here aimed to find the role of follow-up esophagogram and esophagoscopy in detecting anastomotic stricture (AS) in the early stage and to find whether these help in reducing the morbidity in these patients.
Materials and Methods
Neonates with EA Type 3, who were successfully managed with primary anastomosis were prospectively enrolled and followed up in the study. Irrespective of symptoms, contrast esophagography and esophagoscopy were performed at the age of 2–4 months. Those with AS on endoscopy were followed for the need of dilatations required, respiratory complications, and failure to thrive if any.
Results
Out of 64 patients, 32 patients could be followed up till the completion of the study. An esophagogram conducted at 2 months of age revealed significant dye retention in 10 patients, esophageal narrowing in two patients, and normal results in the remaining 18 patients. Endoscopy confirmed AS in 11 out of 32 (34.4%). Preendoscopy, 44% of patients exhibited respiratory morbidity. After endoscopic dilation, none of the patients developed pneumonia requiring admission. Failure to thrive was noted in 9% of the cases.
Conclusion
The use of esophagograms and endoscopies during the healing phase of anastomosis not only aids in the detection of ASs during its formation but also eases the process of their management.
Özofagus Anatomisi ve Histolojisi Orhan BEGER Özofagus Fizyolojisi Bülent SÖNMEZ Özofagus Embriyolojisi Tuba OKYAY Özofagus Hastalıklarında Tanı Yöntemleri Heybet SEMUR Konjenital Özofagus Atrezisi Burhan BEGER Özofagusun Diğer Konjenital Anomalileri Metin ŞİMŞEK Özofagus Motilite Bozuklukları Hüseyin AKDENİZ Akalazya ve Cerrahi Yönetimi Ozan OKYAY Gastro-Özofageal Reflü Hastalığı Abbas ARAS Muhammed Fazıl ARAS Özofagus Kanamaları Mesut AYDIN Özofagus Divertiküler Hastalıkları Özkan YILMAZ İskan ÇALLI Özofageal Herniler Özgür KEMİK Akif AYDIN Özofagus Perforasyonları Musluh HAKSEVEN Özofagus Kostik Yaralanmaları Baran Serdar KIZILYILDIZ Özofagus Yabancı Cisimleri Duygu MERGAN İLİKLERDEN Özofagus Benign Tümörleri Öztekin ÇIKMAN Nuri Emrah GÖRET Ömer Faruk ÖZKAN Özofagus Sarkomları Lokman SOYSAL Özofagus Kistik Lezyonları ve Trakeo-Özofageal Fistüller Ümit Haluk İLİKLERDEN Özofagus Hastalıklarında Anestezi Uygulamaları Hilmi DEMİRKIRAN Arzu Esen TEKELİ Nurçin GÜLHAŞ Özofagus Malign Tümörleri Tolga KALAYCI Özofagus Kanseri Cerrahisi ve Özofageal Rekonstrüksiyon Yöntemleri Mehmet Çetin KOTAN Ümit Haluk İLİKLERDEN Tolga KALAYCI Özofagus Kanserinde Cerrahi Dışı Tedavi Yöntemleri Süleyman ŞAHİN
Background:
Anastomotic strictures (AS) after esophageal atresia (EA) repair are common. While most respond to endoscopic therapy, some become refractory and require surgical intervention, for which the outcomes are not well established.
Methods:
All EA children with AS who were treated surgically at two institutions (2011-2022) were retrospectively reviewed. Surgical repair was performed for those with AS that were either refractory to endoscopic therapy or clinically symptomatic and undergoing surgery for another indication. Anastomotic leak, need for repeat stricture resection, and esophageal replacement were considered poor outcomes.
Results:
139 patients (median age: 12 months, range 1.5 months-20 years; median weight: 8.1 kg) underwent 148 anastomotic stricture repairs (100 refractory, 48 non-refractory) in the form of stricturoplasty (n = 43), segmental stricture resection with primary anastomosis (n = 96), or stricture resection with a delayed anastomosis after traction-induced lengthening (n = 9). With a median follow-up of 38 months, most children (92%) preserved their esophagus, and the majority (83%) of stricture repairs were free of poor outcomes. Only one anastomotic leak occurred in a non-refractory stricture. Of the refractory stricture repairs (n = 100), 10% developed a leak, 9% required repeat stricture resection, and 13% required esophageal replacement. On multivariable analysis, significant risk factors for any type of poor outcome included anastomotic leak, stricture length, hiatal hernia, and patient's weight.
Conclusions:
Surgery for refractory AS is associated with inherent yet low morbidity and high rates of esophageal preservation. Surgical repair of non-refractory symptomatic AS at the time of another thoracic operation is associated with excellent outcomes.
Level of evidence:
Level III.
Introduction: Recurrent tracheoesophageal fistula (RTEF) after surgical repair of esophageal atresia remains a serious complication. Open surgical repair for RTEF is associated with high levels of postoperative morbidity, mortality and repeat recurrence. Although endoscopic management has been expected to be an alternative to re-surgery, the optimal endoscopic management of RTEF remains to be determined. This report describes a successful experience of argon plasma coagulation (APC) for the closure of an intractable RTEF. Case presentation: A newborn male diagnosed as having esophageal atresia with tracheoesophageal fistula (TEF) underwent a primary surgical repair. At 17 months of age, an oral contrast study showed the recurrence of the TEF, and subsequently he underwent a second open surgery. However, a postoperative esophagogram revealed a second recurrence of the TEF. Then, endoscopic treatment options were considered and APC was employed to coagulate the fistula epithelium. At 31 months of age, APC followed by fibrin glue application was performed. By this procedure the RTEF could be occluded safely and successfully without any complications. Conclusion: APC is a simple, safe and effective procedure, and is therefore worth trying as a first attempt for the treatment of RTEF.
Esophageal atresia (EA) is one of the most common, life-threatening congenital malformations of the newborn. Today, survival rates are around 95% and mortality is mainly related to extreme prematurity or severe associated malformations, predominately cardiac anomalies. Operative reconstruction of the esophagus or, in cases of long distance between the esophageal segments, replacement with other organs (stomach, colon, jejunum) is the surgical option. A large variety of operative strategies have been discussed in the past to achieve continuity from the mouth to the stomach. Short-term complications are strictures of the anastomotic region and tracheomalacia. Long-term problems are insufficient propulsive peristalsis, gastric reflux into the esophagus, leading to esophagitis and/or recurrent aspiration. Despite these long-term problems, the overall health-related quality of life in adults is good. However, long-term follow-up studies into adulthood, including esophageal function studies, are needed.KeywordsEsophageal atresiaAssociate malformationsPrimary anastomosisColon interpositionGastric pull-upJejunum interpositionLong-gap problemRecurrent fistulaAnastomotic strictureDysmotilityGastroesophageal refluxTracheomalaciaFollow-up
Objective The aim of this study was to assess incidence, risk factors, and outcomes of distal recurrent tracheoesophageal fistula (RTEF) after repair of esophageal atresia (EA).
Methods Ethical consent was obtained. Data collection was done by review of hospital records of 286 patients (268 in-house and 18 referred) who underwent repair of type C or D EA from 1980 to 2021. Spitz class, long-gap (tracheoesophageal fistula at carina), fundoplication, leakage, and stricture were assessed as RTEF risk factors. Outcome measures were long-term closure of RTEF, retainment of native esophagus, and survival.
Results RTEF occurred in 23 patients (19 in-house) with type C (n = 22) or type D (n = 1) EA with median 4.4 (interquartile range [IQR]: 1.7–13) months after repair. Five patients had late RTEF 3.5 to 16 years after repair. Nineteen (7.3%) in-house patients developed RTEF. Presenting symptoms, age at diagnosis, and presence of anastomotic stricture (AS) are listed. No statistically significant risk factors were found, risk ratio of 0.5 to 2.7 (IQR: 0.1–8.7), p-value of 0.25 to 0.75. Detached closure clip and esophageal foreign body contributed to RTEF in two patients. Sixteen patients underwent rethoracotomy for closure of RTEF, 5 (22%) with AS eventually underwent esophageal reconstruction after a period with cervical esophagostomy. Two patients with late-manifested RTEFs underwent closure with laser cauterization. In 22 patients, treatment of RTEF succeeded, whereas 1 (4%) premature patient died of instant re-RTEF.
Conclusion RTEF had an incidence of 7% with diverse patterns of manifestation and predicting factors. Closure rate and patient survival were excellent, but RTEF with AS predicted loss of native esophagus.
Esophageal atresia with or without tracheoesophageal fistula and tracheobronchomalacia encompass 2 of the most common complex congenital intrathoracic anomalies. Tailoring interventions to address the constellation of problems present in each patient is essential. Due to advances in neonatology, anesthesia, pulmonary, gastroenterology, nutrition and surgery care for patients with complex congenital tracheoesophageal disorders has improved dramatically. Treatment strategies tailored to the individual patient needs are best implimented under the aegis of a comprehensive longitudinal multidisciplinary care team.
Repository-esophageal fistula (REF) in children includes congenital or acquired tracheoesophageal fistula (TEF) and pleuro-esophageal fistula (PEF). TEF is a well-known congenital anomaly that is managed surgically. Recurrent tracheoesophageal fistula (rTEF) occurring after surgical repair of TEF is not an uncommon complication and most of the time requires repeat surgery.
The aim of this paper is to report the outcomes of endoscopic closure of REF in children in Oman. This is a retrospective case series describing the endoscopic closure of REF in children in the Royal Hospital (RH), Oman. Five cases were identified with one of them having acquired PEF while the rest had rTEF. All children had esophageal endoscopic closure of the esophageal fistula using endoclips, cauterization, and glue injection. The patient who had PEF had successful closure of the fistula and only one out of four with rTEF had successful endoscopic closure.
Esophageal endoscopic approach is unsatisfactory in the closure of rTEF but could be effective in the closure of inflammatory PEF. An esophageal approach for the closure of rTEF may need to be consolidated with simultaneous bronchoscopic closure.
Background
Esophageal pulmonary fistula is a special type of acquired tracheoesophageal fistula that occurs after esophageal atresia/tracheoesophageal fistula repair. Thoracotomy is the surgical repair method currently in use, but postoperative outcomes are unclear. Therefore, we aimed to explore the preliminary safety, effectiveness, and feasibility of thoracoscopic surgical repair of esophageal pulmonary fistula.
Methods
We retrospectively collected data from all patients with esophageal atresia/tracheoesophageal fistula at Beijing Children's Hospital from January 2017 to October 2021, and the clinical characteristics of patients with esophageal pulmonary fistula were analyzed. Clinical information was recorded, and follow-up was performed.
Results
Seven patients (five boys and two girls) were diagnosed as esophageal pulmonary fistula. All patients underwent multiple esophageal surgeries and had esophageal strictures before surgical repair. Clinical manifestations included cough, expectoration, and recurrent pneumonia. Esophagography indicated the location of the fistula with a 100% positive rate, while the positive rate of flexible bronchoscopy and chest computed tomography was 57% (4/7) and 43% (3/7), respectively. Surgical repair was achieved using thoracoscopy with an average operation time of 172 min. All patients developed esophageal strictures, four of which had refractory esophageal strictures and underwent esophageal dilations ranged from 5 to 56 times before this surgery, but anastomotic leakage or acquired esophageal pulmonary fistulas were absent post-surgery. After a median follow-up of 22 months, all patients survived, and the symptoms were well controlled.
Conclusions
Esophageal pulmonary fistula is a rare complication of atresia/tracheoesophageal fistula repair. Thoracoscopic surgery is still possible even after previous multiple surgeries in the chest with significant complications and satisfactory results can be achieved in the short term.
Zusammenfassung
Die Ösophagusatresie (ÖA) ist mit einer Inzidenz von 1 : 3000 bis 1 : 4000 Neugeborenen eine seltene, angeborene Erkrankung. Die Therapie beinhaltet obligatorisch die operative Korrektur. Behandlungskonzepte sind abhängig von der Variabilität der ösophagotrachealen Fistelmöglichkeiten, der variablen Distanz der Ösophagussegmente und der prognostischen Relevanz assoziierter Fehlbildungen. Die postoperativen Resultate werden entscheidend durch krankheitsspezifische Symptome und Komplikationen beeinflusst. Ein interdisziplinäres Betreuungskonzept von der pränatalen Diagnostik über die Geburt, die neonatalogische Intensivpflege und kinderchirurgische Versorgung mit hochspezialisierter Kinderanästhesie bietet in einem spezialisierten Kinderzentrum mit Logopädie, Gastroenterologie und Pulmologie die besten Voraussetzungen für eine optimale Lebensqualität. Systematische Nachsorge- und Transitionsprogramme sind für betroffene Kinder essenziell.
Background
A recurrent tracheo-esophageal fistula can complicate esophageal atresia (EA) and tracheo-esophageal fistula (TEF) repair in children. Therapeutic approaches and the rate of recurrence vary widely. Most reports are limited by small cohorts and short-term follow-up, and rates of re-recurrence are significant, making it difficult to select the treatment of choice. We aimed to review our experience with the treatment of recurrent TEF utilizing posterior tracheopexy, focusing on operative risks and long-term outcomes.
Study Design
Retrospective review of EA-TEF patients with recurrent TEF treated at two institutions from 2011-2020. We approach recurrent TEFs surgically. Once the TEF is divided and repaired, the membranous trachea is sutured to the anterior longitudinal ligament of the spine (posterior tracheopexy, PT) and the esophagus is rotated into the right chest (rotational esophagoplasty, RE), widely separating the suture lines. To detect re-recurrence, patients undergo endoscopic surveillance during follow-up.
Results
Sixty-two patients with a recurrent TEF were surgically treated (PT/RE) at a median age of 14 months. All had significant respiratory symptoms. Upon referral, 24 had prior failed endoscopic and/or surgical attempts at repair. Twenty-nine required a concomitant esophageal anastomotic stricturoplasty or stricture resection. Postoperative morbidity included three esophageal leaks, and one transient vocal cord dysfunction. We have identified no recurrences with a median follow-up of 2.5 years, and all symptoms have resolved.
Conclusion
The surgical treatment of recurrent TEFs that incorporates a posterior tracheopexy and rotational esophagoplasty is highly effective at preventing re-recurrence with low perioperative morbidity.
Background & aims
: Recurrent tracheoesophageal fistula (rTEF) after esophageal atresia requires complex management across different specialties. This study reviews our experience and discusses a multidisciplinary (MDT) approach adopted in the past 4 years.
Methods
: We reviewed the medical records of 100 patients with rTEF managed by an MDT approach (post-MDT group) from 2016 to 2019. These cases were compared to a historical group of 35 patients with rTEF from 2012 to 2015 (pre-MDT group).
Results
: Of the 135 patients with rTEF, 124 were referred from other hospitals. Preoperative examination found tracheomalacia in 23 patients, vocal fold immobility in 19 patients, and laryngomalacia in five patients. The incidence of postoperative anastomotic leak, anastomotic stricture, and repeat recurrences was 28.1%, 23.0%, and 8.9%, respectively. The overall mortality rate was 4.4%. No statistical difference in postoperative complications was noted between the two groups. The duration of stay in the pediatric intensive care unit (P = 0.038), the duration of intubation (P = 0.049), the postoperative hospital stay (P = 0.011), and the total length of hospital stay (P = 0.001) were significantly lower in the post-MDT group. Mid-term follow-up showed 23 patients had pathological gastroesophageal reflux. Five of them underwent fundoplication and recovered.
Conclusion
: The MDT approach by fostering coordination of surgical, medical, radiological, and nutritional management is beneficial in the management of rTEF and leads to a satisfactory outcome.
Resumen
Las anomalías congénitas del esófago son malformaciones que se producen durante el desarrollo del árbol traqueobronquial a partir del asa intestinal primitiva durante el período de embriogénesis. La patología de estas malformaciones se conoce cada vez mejor, gracias a experimentos con animales que han permitido crear modelos animales alterando su embriogénesis, pero aún no hay ninguna hipótesis embriológica aceptada de forma unánime. Se han descrito cinco tipos de anomalías: las atresias del esófago, que son las más conocidas y las más frecuentes; las fístulas traqueoesofágicas sin atresia, denominadas en «H»; las estenosis esofágicas que en ocasiones son difíciles de distinguir de las estenosis pépticas secundarias a un reflujo gastroesofágico; las duplicaciones esofágicas congénitas y, por último, los bronquios esofágicos. Todas estas anomalías pueden asociarse a otras malformaciones, como las que aparecen durante el período de embriogénesis. Todas ellas pueden formar parte del síndrome VACTERL. Algunas de estas anomalías, como las atresias del esófago de tipo I o las duplicaciones quísticas del esófago se pueden detectar antes de nacer, pero la gran mayoría de ellas se manifiestan después del nacimiento. La detección de la atresia de esófago debe ser sistemática, debido a las consecuencias graves que puede causar un retraso diagnóstico sobre la salud de los recién nacidos. Los otros diagnósticos son difíciles de establecer en ocasiones, debido a la rareza de estas anomalías. Por todos estos motivos, es indispensable realizar un estudio exhaustivo en cualquier paciente que presente una sintomatología respiratoria repetitiva o crónica, pero también una disfagia no justificada. El manejo y el tratamiento de estas anomalías son muy diversos, dependiendo del tipo de anomalía, pero en la mayoría de los casos la supervivencia y la evolución a largo plazo suelen ser buenas.
Tracheoesophageal fistula (TEF) with esophageal atresia (EA) is a common congenital anomaly that is associated with significant respiratory morbidity throughout life. The objective of this document is to provide a framework for the diagnosis and management of the respiratory complications that are associated with the condition. As there are no randomized controlled studies on the subject, a group of experts used a modification of the Rand Appropriateness Method to describe the various aspects of the condition in terms of their relative importance, and to rate the available diagnostic methods and therapeutic interventions on the basis of their appropriateness and necessity. Specific recommendations were formulated and reported as Level A, B, C based on whether they were based on “strong”, “moderate” or “weak” agreement. The tracheomalacia that exists in the site of the fistula was considered the main abnormality that predisposes to all other respiratory complications due to airway collapse and impaired clearance of secretions. Aspiration due to impaired airway protection reflexes is the main underlying contributing mechanism. Flexible bronchoscopy is the main diagnostic modality, aided by imaging modalities, especially CT scans of the chest. Non‐invasive positive airway pressure support, surgical techniques such as tracheopexy and rarely tracheostomy are required for the management of severe tracheomalacia. Regular long‐term follow‐up by a multidisciplinary team was considered imperative. Specific templates outlining the elements of the clinical respiratory evaluation according to the patients’ age were also developed.
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Purpose:
The aim of this study was to investigate the clinical effectiveness of azygos vein preservation when using a thoracoscope in the operation for type III esophageal atresia for children.
Materials and Methods:
We retrospectively analyzed the clinical data of 34 patients (group B) who underwent the thoracoscopic operation of type III esophageal atresia with azygos vein preservation from January 2017 to April 2018. The clinical data of 36 patients (group A) who underwent the thoracoscopic operation of type III esophageal atresia with azygos vein mutilation from April 2015 to December 2016 were selected as a control group.
Results:
All patients were successfully operated on. There was no significant difference in the operation time, amount of bleeding, mechanical ventilation time, and intensive care time between the azygos vein mutilation group and azygos vein preservation group. Compared with the operation severing the azygos vein, preservation of azygos vein has obvious advantages in postoperative hospitalization time, the incidence of anastomotic leakage (P < .05). The follow-up results for 1 year after the operation showed that the incidence of anastomotic stricture with preservation of azygos vein was significantly lower than that with azygos vein transection (P < .05).
Conclusions:
The thoracoscopic operation of type III esophageal atresia with azygos vein preservation can not only improve the edema of esophageal tissue around the esophagus, but also reduce the occurrence of esophageal anastomotic leakage and accelerate the postoperative recovery of children, and it has the same safety as the operation with azygos vein mutilation.
Aims
To illustrate the construction of statistical control charts and show their potential application to analysis of outcomes in children's surgery.
Patients and methods
Two datasets recording outcomes following esophageal atresia repair and intestinal resection for Crohn's disease maintained by the author were used to construct four types of charts. The effects of altering the target signal, the alarm signal and the limits are illustrated. The dilemmas in choice of target rate are described. Simulated data illustrate the advantages over hypothesis testing.
Results
The charts show the author’s institutional leak rate for esophageal atresia repair may be within acceptable limits, but that this is dependent on the target set. The desirable target is contentious. The leak rate for anastomoses following intestinal resection for Crohn's disease leak is also within acceptable limits when compared to published experience, but may be deteriorating. The charts are able to detect deteriorating levels of performance well before hypothesis testing would suggest a systematic problem with outcomes.
Conclusions
Statistical process control charts can provide surgeons with early warning of systematic poor performance. They are robust to volume–outcome influences, since the outcome is tested sequentially after each procedure or patient. They have application in a specialty with low frequencies of operations such as children's surgery.
Type of study
Diagnostic test.
Level of evidence
Level II.
Revisional foregut surgery for the pediatric patient is relatively uncommon when compared to the adult patient population. Early recognition of the presenting signs and symptoms in the neonate, infant, or child may ultimately allow for nonoperative management of unplanned outcomes. Nonetheless, the surgeon ought to be prepared to manage the rare operative complications. The following chapter will detail the six most commonly performed foregut operations for the pediatric population and their respective morbidities requiring revisional surgery.
Introduction Many aspects of the management of esophageal atresia (EA) and tracheoesophageal fistula (TEF) are controversial and the evidence for decision making is limited. Members of the European Reference Network for Rare Inherited Congenital Anomalies (ERNICA) conducted a consensus conference on the surgical management of EA/TEF based on expert opinions referring to the latest literature.
Materials and Methods Nineteen ERNICA representatives from nine European countries participated in the conference. The conference was prepared by item generation, item prioritization by online survey, formulation of a final list containing the domains diagnostics, preoperative, operative, and postoperative management, and literature review. The 2-day conference was held in Berlin in October 2018. Anonymous voting was conducted via an internet-based system. Consensus was defined when 75% of the votes scored 6 to 9.
Results Fifty-two items were generated with 116 relevant articles of which five studies (4.3%) were assigned as level-1evidence. Complete consensus (100%) was achieved on 20 items (38%), such as TEF closure by transfixing suture, esophageal anastomosis by interrupted sutures, and initiation of feeding 24 hours postoperatively. Consensus ≥75% was achieved on 37 items (71%), such as routine insertion of transanastomotic tube or maximum duration of thoracoscopy of 3 hours. Thirteen items (25%) were controversial (range of scores, 1–9). Eight of these (62%) did not reach consensus.
Conclusion Participants of the conference reached significant consensus on the management of patients with EA/TEF. The consensus may facilitate standardization and development of generally accepted guidelines. The conference methodology may serve as a blueprint for further conferences on the management of congenital malformations in pediatric surgery.
We analyzed retrospectively the outcomes in long gap Gross type C esophageal atresia. We hypothesized that outcomes in type C (long gap) atresia differ from type C (normal gap) and be similar with outcomes in Gross type A and B esophageal atresia. Location of the distal tracheoesophageal fistula (TEF) at the carina was chosen as the hallmark of type C atresia (long gap). We compared the type of esophageal repair, major reoperations for anastomotic complications and gastroesophageal reflux, and long-term mucosal changes between type C (normal gap), type C (long gap), and type A/B. We analyzed the hospital charts of 247 successive patients from 1984 to 2014 who either underwent repair of esophageal atresia in our institution (n = 232) or were referred from elsewhere because of anastomotic complications (n = 15). Among the 232 patients of our institution, 181 had type C and 21 type A or B esophageal atresia. Twenty-two (12%) of type C patients had TEF at the carina and were considered as type C (long gap). The referred patients included a disproportionately high number (42%) of patients with type C (long gap). We attempted primary anastomosis in 98% of patients with type C (normal gap), in 95% with type C (long gap), and 53% with type A/B underwent delayed primary anastomosis. Leakage after primary anastomosis occurred in 40% of patients with type A/B and in 23% with type C (long gap) compared with 6% in patients with type C (normal gap) (P < 0.05). Recalcitrant anastomotic stricture that eventually required esophageal resection occurred in 30% of patients with type A/B and in 18% with type C (long gap) compared with 3% in patients with type C (normal gap) (P < 0.05). The overall rate of major reoperations for anastomotic complications after primary anastomosis, type A/B (36%), type C (long gap) (27%), and antireflux surgery, type A/B (100%) and type C (long gap) (61%) were higher than in type C (normal gap), (9% and 24%), (P < 0.05 in both). Ten (47%) patients with type A/B esophageal atresia (primary anastomosis not possible n = 10), three (14%) with type C (long gap) (primary anastomosis not possible n = 1, significant loss of esophageal length after complications n = 2) and two (1%) with type C (normal gap) (significant loss of esophageal length after complications n = 2) underwent esophageal reconstruction. Endoscopic follow-up, median length 7.0 (IQR: 3.0–14) years, disclosed gastric metaplasia in 31% and 33% of patients with type A/B and type C (long gap) compared with 11% in type C (normal gap) (P < 0.05). Intestinal metaplasia was found in one patient type C (normal gap) (0.7%) and one with type C (long gap) (5.6%), (P = 0.21), only. The outcomes of type C (long gap) esophageal atresia are associated with more frequent complications, gastroesophageal reflux and esophageal mucosal changes than outcomes in type C (normal gap). Outcomes in type C (long gap) esophageal atresia resemble those in type A/B. The percentage of patients who remain with their native esophagus is, however, higher in type C (long gap) atresia (86%) than in type A/B (53%).
Background/purpose:
In a sparsely investigated field, we aimed to evaluate the use of special preschool/school support among children with repaired esophageal atresia (EA) and/or tracheoesophageal fistula (TEF), the predicting clinical factors for this support, and level of school absence.
Methods:
Data on 119 EA/TEF children 2-17years old were collected through medical records and questionnaires (response rate 95%). Logistical regression analysis identified clinical predictors of special preschool/school support in the population without genetic disorders (n=105). Nominal hypothesis testing was performed using Fisher's exact test (p<0.05).
Results:
Of the 119 children, 35.3% received special preschool/school support; 26.8% educational support, 21.8% support with nutritional intake issues and 13.4% received both types of support. Educational support was independently predicted by birth weight<2500g (p=0.026) and associated anomalies (p=0.049), nutritional intake support by gastrostomy insertion (p=0.0028), and both types of supports by major revisional surgery (p=0.0081). School absence ≥1month/year, present in 25.5% of the children, was more frequently reported in children receiving preschool/school support, in preschoolers and in those with persistent respiratory problems (p<0.05).
Conclusions:
Special preschool/school support is provided for approximately one-third of EA/TEF children. In EA/TEF children without genetic disorders, use of this support is predicted by congenital and surgical factors, and related to frequent school absence.
Recurrent tracheoesophageal fistula (rTEF) is a complex complication after the repair of esophageal atresia (EA) and remains a challenge because of difficulties in preoperative management and the substantial rates of mortality and morbidity after reoperation. By reviewing a single institution's experience in the management of rTEF and assessing the outcome, we aimed to provide an optimal approach for managing rTEF and to evaluate growth and feeding problems after reoperations. The medical records of 35 patients with rTEF treated at a single institution from June 2012 to December 2015 were reviewed, and follow-up data were collected from all survivors. The diagnosis of rTEF was made using a modified esophagram in 32 of 35 patients. Before reoperation, all patients received continuous aspiration to prevent reflux and aspiration pneumonia by placing two nasogastric tubes at the level of the fistula and into the stomach, and they received enteral nutrition through a jejunal feeding tube. Thirty-five patients received a total of 41 reoperations, including 12 operations of fistula resection, 28 reanastomosis, and 1 esophageal replacement. The incidence of postoperative anastomotic leak (AL), anastomotic stricture (AS), and repeat recurrences was 40.0%, 17.1%, and 11.4%, respectively. The mortality rate was 8.6%. All survivors achieved full oral intake. Mid-term follow-up (median of 18 months) revealed that 7 (21.9%) presented prolonged meal time, 6 (18.8%) had feeding refusal, 8 (25.0%) experienced coughing during feeding, and 7 (21.9%) had vomiting during feeding. According to the growth data, 5 survivors (15.6%) presented with growth retardation, including stunting (n = 1), wasting (n = 2), and underweight (n = 2). The modified esophagram is an effective and reliable method for diagnosing rTEF. Optimized preoperative management and surgical techniques lead to a satisfactory outcome. Nevertheless, nutritional evaluation and feeding guidance by a nutritionist after reoperation are recommended.
The usual indications for oesophageal replacement in childhood are intractable corrosive strictures and long-gap oesophageal atresia. Generally, paediatric surgeons attempt to preserve the native oesophagus with repeat dilatations. However, when this is not successful, an appropriate conduit must be fashioned to replace the oesophagus. The neo-oesophagus should allow normal oral feeding, not have gastro-oesophageal reflux, and be able to function well for the life-time of the patient.
A Medline search for oesophageal replacement, oesophageal atresia, gastric transposition, colon transposition, gastric tube, caustic stricture was conducted. The commonest conduits including whole stomach, gastric tube, colon and jejunum are all discussed.
No randomised controlled studies exist comparing the different types of conduits available for children. The techniques used tend to be based on personal preference and local experience rather than on any discernible objective data. The biggest series with long-term outcome are reported for gastric transposition and colon replacement. Comparison of a number of studies shows no significant difference in early or late complications. Early operative complications include graft necrosis, anastomotic leaks and sepsis. Late problems include strictures, poor feeding, gastro-oesophageal reflux, tortuosity of the graft and the development of Barrett's oesophagus. The biggest series, however, seem to have lower complications than small series probably reflecting the experience, built up over years, in their respective centres.
Long-term follow-up is recommended because of the risks of late strictures, excessive tortuosity of the neo-oesophagus and the development of Barrett's oesophagus.
Background
We investigated whether removable stents, such as self-expandable plastic stents (SEPSs) and fully covered self-expandable metal stents (FCSEMSs) could provide an alternative treatment for recalcitrant strictures and esophageal perforations after esophageal atresia (EA) repair.
Objective
The primary aim of our study was to evaluate technical feasibility. Secondary aims were to evaluate safety and procedural success.
Design
Retrospective study.
Setting
Tertiary-care referral center.
Patients
A total of 24 children with EA.
Interventions
Retrospective review of all children with EA who underwent dilation and esophageal stent placement from January 2010 to February 2013 at our institution.
Main Outcome Measurements
Healing of perforation and stricture resolution at 30 and 90 days.
Results
A total of 41 stents (SEPSs 14, FCSEMSs 27) were placed in 24 patients with EA during the study period, including 14 who had developed esophageal leaks. Procedural success of esophageal stent placement in the treatment of refractory strictures was 39% at 30 days and 26% at 90 days. The success rate was 80% for closure of esophageal perforations with stent therapy after dilation and 25% for perforations associated with surgical repair. Adverse events of stent placement included migration (21% of SEPSs and 7% of FCSEMSs), granulation tissue (37% of FCSEMSs), and deep ulcerations (22% of FCSEMSs).
Limitations
Retrospective study with small sample size.
Conclusion
SEPSs and FCSEMSs can be placed successfully in small infants and children with a history of EA repair. The stents appear to be safe and beneficial in closing esophageal perforations, especially post-dilation. However, a high stricture recurrence rate after stent removal may limit their usefulness in treating recalcitrant esophageal anastomotic strictures.
This article presents a 30-year review of 38 recurrent tracheoesophageal fistulas. The initial 26 cases were presented in 2009 at the annual meeting of the British Association of Pediatric Surgeons and the European Association of Pediatric Surgeons Joint Conference and published in the Journal of Pediatric Surgery (Bruchet al. J Pediatr Surg 45:337-340, 2010). In the initial cohort of 26 patients, 18 had a leak after their primary operation and 22 had respiratory symptoms leading to the discovery of the recurrent fistula. The diagnosis was made by a contrast study in 24. The repairs entailed replacing a catheter through the fistula, separating the trachea and esophagus completely using sharp dissection and placing vascularized tissue, either pleura or pericardium between the suture lines. Postoperative complications included seven anastomotic leaks, four strictures and three recurrent fistulas. Long-term follow-up (median of 84 months) showed that 21 took all of their nutrition by mouth, three were tube fed and two required a combination of both. Of the 23 patients with growth chart data, 16 fell into the first quartile of the growth chart, whereas none fell between the 75th and 100th percentile. In conclusion, this initial series of 26 patients along with the updated additional series of 12 patients is the largest series thus far reported in the literature. All 38 patients represent the characteristics of recurrent tracheoesophageal fistulas, including techniques to make the diagnosis and to provide a secure closure of the fistula, and the long-term outcomes of these patients.
The aim of this study was assessment of the contemporary outcomes of oesophageal atresia (OA) in a national paediatric surgical centre.
A review of the hospital records of all patients who underwent repair of oesophageal atresia (OA) in our institute between 1991 and 2011 was performed.
The study included 130 consecutive infants with OA: types A (n=4, 3%), B (n=2, 2%), C (n=110, 85%), D (n=5, 4%), and E (n=9, 7%). Median follow-up was 8.8 (range 0.1-21) years. Twenty-nine (22%) infants had cardiac and 76 (58%) other anomalies, and seventeen (13%) had a long-gap OA. The final repair was primary (n =113, 87%) or delayed (n=3) end-to-end anastomosis, oesophageal replacement (n=8) (6%) with gastric tube (n=4) or with jejunum interposition (n=4), and closure of the trachea-oesophageal fistula (Type E, n=9). Oesophageal continuity was achieved in all patients. Overall mortality was 3/130 (2%) and caused by gastric perforation (n=1), prolonged apnoeic spell (n =1), and food asphyxiation (n=1). Oral feeds were achieved in 121 (94%) children. Eight (6%) children remain dependent on feeding ostomy. Long-gap OA was a major predictor of post-repair complications.
The modern outcome for infants with OA is characterized by an extremely low hospital mortality and satisfactory oesophageal function, enabling full oral feeds in the vast majority of children.
Thoracoscopic approach for repair of esophageal atresia (EA) and tracheo-esophageal fistula (TEF) has become a standard procedure in many pediatric surgical centers. Thoracoscopic surgery in a newborn is demanding from both the surgeon and the patient. The potential benefits for the newborn are still discussed by neonatologists, pediatric intensive care physicians, and also parents. The aim of our investigation was to clearly define perioperative outcome and complication rates in children undergoing thoracoscopic versus open surgery for EA and TEF repair.
We reviewed the records of 68 newborns undergoing surgery for EA and TEF between March 2002 and February 2010. Patient data of open versus thoracoscopic approach were compared regarding operating time, intraoperative as well as postoperative pCO(2)max values, postoperative ventilation time, and complications. Specific patient data are reported with the median and range. Data analysis was done with the JMP(®) 7.0.2 statistical software (SAS Institute, Cary, NC).
For the 68 patients, the mean gestational age was 35 weeks (28-41), the median birth weight was 2720 g (1500-3510 g) in the thoracoscopic group and 2090 g (780-3340 g) in the open group. There were 36 girls and 32 boys. Thirty-two children had associated anomalies. Twenty-five children were undergoing a thoracoscopic procedure. In 8 cases, the operation was converted to open thoracotomy. Another 32 children received a thoracotomy. In 11 newborns, a cervical esophagostomy was performed because of long-gap EA and these patients were excluded from the study. Operating time was 141 minutes (77-201 minutes) in the thoracoscopic group and 106 minutes (48-264 minutes) in the thoracotomy group, with significant difference (P=.014). Values of pCO(2)max during operation were 62 mm Hg (34-101 mm Hg) in the thoracoscopic group and 48 mm Hg (28-89 mm Hg) in the open group, with significant difference (P=.014). Postoperative ventilation time was 3 days (1-51 days) in all groups, with no significant difference (P=.79). Early complications were noticed in 9 children undergoing thoracoscopy and in 8 patients of the thoracotomy group, again with no significant difference (P>.05).
Thoracoscopic repair of EA with TEF is justified because of a comparable perioperative outcome to open surgery, competitive operating times, decreased trauma to the thoracic cavity, and improved cosmesis despite skeptical considerations. Complication rates are not higher than in children operated on through a thoracotomy. However, a learning curve has to be taken into account and large experience in minimal invasive surgery is mandatory for this procedure. Larger series have to be expected for a more objective evaluation of perioperative as well as long-term outcomes. To our opinion, the thoracoscopic approach appears to be favorable and could be a future standard.
Strictures of the esophagus in children may have multiple etiologies including congenital, inflammatory, infectious, caustic ingestion, and gastroesophageal reflux (peptic stricture [PS]). Current literature lacks good data documenting long-term outcomes in children. This makes it difficult to counsel some patients about realistic treatment expectations. The objective of this study is to evaluate our institutional experience and define the natural history and treatment outcomes.
A retrospective review of clinical data obtained from children who underwent dilation for PS was performed.
Over the past 30 years, 114 children and adolescents received 486 dilations. The most common indications for stricture dilation were PS (42%) and esophageal atresia (38%). Other lesser indications included congenital, foreign body, corrosive, cancer, radiation, allergic, and infectious. This review focuses on the 48 children with PS. Of the children with PS, a congenital anomaly was identified in 23 children; and 12 had neurologic impairment. Average age at presentation was 10.2 years (range, 0.5-18.3 years). Most patients had had symptoms for many months before diagnosis. Peptic stricture was most common in the lower esophagus (n = 39). However, middle (n = 8) and upper (n = 1) strictures were occasionally identified. Noncompliance with medical therapy was a challenge in 12% (n = 5) of children. Children with a PS received a median of 3 dilations, but a subset of 5 patients with severe strictures underwent up to 48 dilations (range, 1-48). Repeated dilations were required for a median of 20 months (range, 1-242 months). Among patients receiving esophageal dilation for PS, 94% required an antireflux procedure (19% required a second antireflux surgery). A subgroup of patients (n = 10) was identified who required extended dilations, multiple surgeries, and esophageal resection. This subgroup had a significantly longer period of symptomatic disease and increased risk of esophageal resection compared with those patients requiring fewer dilations. Surgical resection of the esophageal stricture was ultimately required in 3 children with PS after failure of more conservative measures.
Children and adolescents presenting with reflux esophageal stricture (PS) frequently require antireflux surgery, redo antireflux surgery, and multiple dilations for recurrent symptoms. We hope that these data will be of use to the clinician attempting to counsel patients and parents about treatment expectations in this challenging patient population.
The aim of the study is to investigate whether a tracheoesophageal fistula (TEF) found after the primary repair of type C esophageal atresia (EA) is a recannulation of the original fistula, a missed proximal fistula, or other rare foregut malformation.
Between 2000 and 2009, 143 different types of patients with EA were admitted in our hospital. Seven patients (2 from our series, 5 referred to us by other hospitals with the history of primary repair of type C EA) had late presenting TEF. Esophagogram, 3-dimensional computed tomographic (CT) reconstruction, bronchoscopy, and reoperation were performed to confirm the TEF. Their medical records were reviewed and summarized.
Persistent feeding or respiratory problems were the common symptom. The mean age of the first appearance was 17 ± 26 (1-63) months. Preoperative diagnosis was made by esophagograms and bronchoscopy in 6 patients. Reoperations were performed in all patients through thoracotomy. Missed proximal TEF shown as a distinct fistula above the primary anastomosis without much adhesion was confirmed in 5 cases. A recurrent TEF was found in 1 case. A case of communicating bronchopulmonary foregut malformation was confirmed by 3-dimensional CT reconstruction and reoperation.
A missed proximal TEF after repair of EA may be misdiagnosed as a recurrent TEF. Accurate preoperative diagnosis depends on combined evaluations of radiologic contrast study, 3-dimensional CT, and bronchoscopy.
This review provides a blueprint to deal with the diagnosis and management of recurrent tracheoesophageal fistulas.
A retrospective review over 27 years found 26 recurrent tracheoesophageal fistulas. Descriptive statistical analyses were performed.
In this cohort of 26 patients, 18 had a leak after their primary operation; and 22 had respiratory symptoms leading to the discovery of the recurrent fistula. The diagnosis was made by contrast study in 24. The repairs entailed placing a catheter through the fistula; separating the trachea and esophagus using sharp dissection; and placing tissue, preferably pericardium, between the suture lines. Postoperative complications included 7 anastamotic leaks, 4 strictures, and 3 recurrent fistulas. Long-term follow-up (median of 84 months) showed that 21 took all of their nutrition by mouth, 3 were tube fed, and 2 required a combination. Of the 23 patients with growth chart data, 16 fell in the first quartile of the growth chart, whereas none fell between the 75th and 100th percentile.
This series, the largest to date, describes characteristics of recurrent tracheoesophageal fistulas, including techniques to make the diagnosis and provide a secure closure of the fistula, and the long-term outcomes of these patients.
The authors analyzed the outcome for 357 infants with oesophageal atresia and 15 with H-type tracheoesophageal fistula treated from 1980 through 1992. Survival according to Waterston risk categories was 99% for group A, 95% for group B, and 71% for group C. Presently, with optimal management, virtually all infants in groups A and B should survive. When examining the risk factors in the infants who died, two criteria were found to be important predictors of outcome: birth weight of less than 1,500 g and the presence of major congenital cardiac disease. A new classification for predicting outcome in oesophageal atresia is proposed: group I: birth weight > or = 1,500 g, without major cardiac disease, survival 97% (283 of 293); group II: birth weight < 1,500 g, or major cardiac disease, survival 59% (41 of 70); and group III: birth weight < 1,500 g, and major cardiac disease, survival 22% (2 of 9).
Given the bias that the native esophagus is the best conduit between the oropharynx and the stomach, the authors report a "conservative" approach to massive esophageal leak, which may be considered "radical" by others. Major disruption of the anastomosis after primary repair of esophageal atresia is a recognized and feared complication. Historically, management has been the performance of cervical esophagostomy and gastrostomy. The aim of this report is to describe the authors' approach to this difficult and serious complication.
A 15-year retrospective analysis was performed of all patients having esophageal atresia. Data collection focused on the management of all patients with clinically significant esophageal disruption. Radiographically detected (clinically asymptomatic) leaks were managed by continuation of drainage by thoracostomy tubes already in place and are not included. Reoperative thoracotomies were performed, which included primary repair (2), placement of pleural patch alone (2), pleural patch with intercostal muscle flap buttress (2), and operative debridement and drainage alone (1).
It was noted that seven patients had clinically significant esophageal disruption requiring reoperation, with circumferential disruptions ranging from 15% to 85%. Presentation included persistent pleural collection (4) and pneumothorax (3). Both patients who underwent primary repair had no evidence of leakage on follow-up esophagograms, neither did one with a pleural patch alone and one with an intercostal muscle flap. Five of the seven patients were tolerating oral feedings at the time of follow-up (range, 6 months to 8 years). One of the two others (both currently inpatients), has a recurrent leak associated with mediastinitis, and the other (who had primary repair) has a presumed neurological impairment of eating.
Clinically significant disruption of primary esophageal repair should not warrant a cervical esophagostomy and placement of a gastrostomy tube, thus precluding eventual use of the native esophagus. The authors have shown that management by reoperation with primary repair, intercostal muscle flap with or without pleural patch, and/or drainage allows the patient to maintain the native esophagus and yields a generally good outcome after a prolonged healing time.
Esophageal replacement is associated with significant morbidity that may lead to operative interventions. This study reviews the management and outcome of children who underwent reoperation after esophageal replacement.
Eighteen patients who underwent esophageal replacement from 1985 to 1997 were reviewed retrospectively. Ten patients underwent reoperation. Patient management, perioperative morbidity, and the dietary intake at follow-up were recorded for each patient.
Of the reoperated patients, 7 had esophageal atresia, 2 had caustic ingestion, and 1 had achalasia. Nine patients received a colon interposition, and 1 received a reverse gastric tube as the initial esophageal replacement. Seven patients required revision of the anastomoses. Three patients required complex esophageal reconstruction: 1 underwent gastric transposition, 1 underwent free jejunal graft, and 1 underwent gastric transposition combined with free jejunal graft. Seven patients were eating well at follow-up. Two patients still required partial gastrostomy tube feeding. One patient died 6 months postoperatively from aspiration pneumonia.
Esophageal replacement continues to be a challenging operation associated with significant complications. Most reoperative procedures were directed toward strictures and persistent fistulae. Complete graft failure can be managed by gastric transposition or free jejunal graft. Despite the perioperative morbidity, most patients have good functional outcome.
Reoperative esophageal surgery can be a very challenging endeavor. Preoperative evaluation, planning and preparation are essential to optimize results. A general reoperative approach and the range of reconstructive options are outlined. Management of specific problems is discussed including stricture, recurrent gastroesophageal reflux, recurrent tracheoesophageal fistula, esophageal interposition, and recurrent achalasia.
Reconstruction of the esophagus in children remains a challenge. Although jejunal grafts retain peristaltic activity, large series with long-term follow up are rare. We like to present our experience in a series of 24 children.
In the period 1988 through 2005, 24 children received an orthotopic jejunal pedicle graft reconstruction of the esophagus. Nineteen had esophageal atresia (18 had no distal fistula; all but 1 had a jejunal graft as a primary procedure), 3 had an extensive caustic stricture, and 2 had a peptic stricture. All strictures had been dilated many times, and peptic strictures had been treated with antireflux surgery as well. Median age at reconstruction was 76 days in the esophageal atresia group. The technique involves a right-sided thoracotomy with preparation of the esophageal ends or resection of the diseased esophagus. At laparotomy, a small pediculated jejunal graft is prepared and placed transhiatally in an orthotopic position in the right chest.
All patients survived, and none of the grafts were lost. There were 5 intrathoracic leaks, 4 in the esophageal atresia group and 1 in peptic stricture group, requiring reoperation in 1. In the esophageal atresia group, there was 1 early distal stenosis requiring reoperation. In patients in which the distal esophagus was preserved (esophageal atresia and caustic stricture group), there were always signs of distal functional subobstruction, responding to dilatation in all but 1 patient. Gastroesophageal reflux was not a problem except for 1 patient with esophageal atresia, in whom the distal esophagus was resected because of ongoing distal obstruction with dilatation of the graft. Most patients eat and grow well, and respiratory problems were rare.
Orthotopic jejunal pedicle graft reconstruction of the esophagus in children is a demanding operation with considerably morbidity but good long-term results.
Benign oesophageal strictures may occur as a complication of caustic ingestion or severe gastro-oesophageal reflux or as a sequela of oesophageal surgery and other fibrosing conditions. The traditional initial treatment of oesophageal strictures is intraluminal dilation; however, even if frequent, this occasionally may not provide adequate oesophageal lumen capacity or give significant symptom-free intervals, and restricturing after dilation is difficult and challenging. Topical postdilation application of an antifibrotic agent, mitomycin-C, in the treatment of an oesophageal stricture has been described.
Eight centres participated, with a total of 16 patients (4 girls), median age 48 (range 0-276) months. The causes of stricture were as follows: caustic (10), post-trachea-oesophageal fistula repair (2), peptic (2), Crohn disease (1), and dystrophic epidermolysis bullosa (1). The median (range) length and diameter of the strictures were as follows: 22 mm (8-50 mm) and 1.5 mm (1-6 mm). Of the 16 patients, 15 had undergone repeated dilations varying from 3 to more than 1000 (daily self-bouginage) before mitomycin-C, and the median interval between dilations was 4 weeks. Mitomycin-C 0.1 mg/mL was applied after dilation for a median time of 3.5 minutes and a median of 3 (1-12) times.
Major success, both endoscopic and clinical improvement or cure, occurred in 10 of 16 patients. In 3 of 16 patients the interval period between dilations increased dramatically. Failure of therapy was considered in 3 of 16. All of the patients remained symptom free for a follow-up time of as long as 5 years.
Postdilation application of topical mitomycin-C resulted in major success in 62.5% of patients and partial success in 19%, and it may be a useful strategy in oesophageal strictures of differing causes that are refractory to repeated perendoscopic dilation.
The surgery of infancy and childhood. Philadelphia: WB Saunders
- Re Gross
Gross RE. The surgery of infancy and childhood. Philadelphia: WB Saunders; 1953.