Review Article
365
Management of Recurrent Tracheoesophageal Fistulas: A Systematic Review Olugbenga Aworanti1,2
Sami Awadalla1
1 Department of Paediatric Surgery, Children’s University Hospital,
Dublin, Ireland 2 Department of Paediatric Surgery, Our Lady’s Children’s Hospital, Crumlin, Dublin, Ireland
Address for correspondence Olugbenga Aworanti, MBBS, MRCSEd, MSc, Department of Paediatric Surgery, Children’s University Hospital, Temple Street, Dublin 1, Ireland (e-mail: [email protected]).
Abstract
Keywords
► recurrent tracheoesophageal fistula ► systematic review ► endoscopic ► management
Introduction The management of a recurrent tracheoesophageal fistula (RTEF) includes either open surgery (OS) or an endoscopic treatment (ET); the ideal option is unclear. We aim to comparatively review all published treatment options, and outcomes, for managing RTEF. Materials and Methods A literature search was performed using the keywords “recurrent tracheoesophageal fistula.” All English language articles describing the management of RTEF in children were reviewed. A synthesis of the relevant data is presented in a descriptive form due to the heterogeneity of the included articles. Results A total of 44 papers between 1955 and 2013 described 165 patients; 57 ET and 108 OS. Of the 57 ET patients, there was an 84% success rate compared with 93.5% of 108 OS patients; the failed ET cases were all successfully treated by OS. The refistulation rate after OS was 21% and an average of 1.1 (range 1–2) procedures were required. After ET, the refistulation rate was 63% and an average of 2.1 (range 1–6) treatments were required for success; these results were reported after a maximum follow-up of 9 years and 23 years for ET and OS, respectively. The major complications after OS were 17 (16%) leaks and 4 (3.7%) deaths, while for ET 3 (5%) suffered respiratory distress postoperatively and there was 1 (1.7%) death. Conclusion OS for RTEF has a low morbidity and mortality, a higher success rate, and requires fewer treatments than an endoscopic repair. The ideal ET is undecided but it remains a viable alternative provided treatment failures are anticipated and prompt redo treatments initiated to prevent ongoing respiratory morbidity.
Introduction The spectrum of esophageal atresia (EA) and tracheoesophageal fistula (TEF) include an isolated EA, EA with a proximal and/or distal TEF and the “H” or “N” type fistula. Since the initial description of the open surgical repair1 and subsequent thoracoscopic approaches2,3 the outcomes have been very encouraging. Anastomotic leaks, esophageal strictures, gastroesophageal reflux, and tracheomalacia are complications
received October 3, 2013 accepted after revision January 11, 2014 published online March 28, 2014
pediatric surgeons are familiar with. The incidence of a fistula recurrence range from 1.9 to 11% after open and thoracoscopic repairs.2,4–8 The diagnosis of these recurrent tracheoesophageal fistulas (RTEF) can often be elusive to the unsuspecting physician and the ideal approach to their management is still in debate. Proponents of an endoscopic approach to this problem frequently state that the morbidity, mortality, and second recurrence rate are too high with an open approach and majority directly or indirectly reference
© 2014 Georg Thieme Verlag KG Stuttgart · New York
DOI http://dx.doi.org/ 10.1055/s-0034-1370780. ISSN 0939-7248.
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Eur J Pediatr Surg 2014;24:365–375.
Management of RTEF: A Systematic Review
Aworanti, Awadalla
the only literature review of open surgery (OS) from 1970 by Kafrouni et al.7 Here, we reviewed the published treatment options, and their outcomes, for managing RTEF. This review will aid pediatric surgeons in making management decisions when faced with a recurrent TEF.
Patients/Materials and Methods A Medline, Embase, and web of science searches were performed using the keywords recurrent tracheoesophageal fistula. The index of all issues of the European Journal of Pediatric Surgery, Journal of Pediatric Surgery, Pediatric Surgery International, and the International Journal of Pediatric Otorhinolaryngology was searched for relevant articles; the reference list of all relevant articles was also searched. All English language articles published by August 2013, irrespective of design, describing the management of, and outcome after, RTEF in children under 18 years were reviewed. The data obtained included number of patients treated, treatment modality employed, effectiveness of each treatment modality, and the outcomes after treatment. These outcomes include the successful single treatments, number of treatments required, number of treatment failures requiring conversion to an alternate treatment modality, and the incidence of postoperative complications. A synthesis of the data is presented in a descriptive form due to the heterogeneity of the included articles.
Results
Some perform a repeat esophagoscopy and bronchoscopy to confirm closure at both ends of the fistula, exclude dislodgement/aspiration of remnant sealant, and exclude intraluminal damage particularly when electrocautery or laser de-epithelialization is performed. The child is usually kept nil per oral for several days and a follow-up contrast esophagram is obtained before oral feeding.
Open Surgical Repair
A total of 23 papers between 1984 and 2012 described 57 patients undergoing some form of endoscopic treatment (ET) while 21 papers between 1955 and 2013 described 108 patients that underwent an open surgical repair (►Tables 1 and 2). A total of 165 cases of RTEF are described and there were 44 (26.7%) cases associated with anastomotic leaks while 35 (21.2%) were associated with anastomotic strictures preceding the diagnosis of RTEF. Below is a summary of the published operative principles.
Endoscopic Treatment This is usually performed via bronchoscopy but can be done via esophagoscopy.9,10 Three categories of ET have been described as follows: • Injection of a sealant into the fistula tract or submucosa. The sealants commonly used are fibrin glue (Tisseel: Baxter Corporation, Ontario, Canada; Tissucol: Baxter, Vienna, Austria; and Tisseal: Baxter Inc., Deerfield, Illinois, United States)11–14 and n-butyl cyanoacrylate (n-BCA, enbucrilate, and Histoacryl: B. Braun Melsungen AG, Mesungen, Germany). • De-epithelialization of the fistula tract involves abrading, denuding, cauterizing, fulgurating, chemocauterizing, sclerosing, or destroying the epithelial lining of the fistula using diarthermy,15,16 Nd:YAG laser,17 electrocautery,17 trichloroacetic acid,18 or potassium titanyl phosphate laser.19 • A combination of de-epithelialization and injection of a sealant: submucosal 30% NaCl (sclerosant) þ n-BCA (sealEuropean Journal of Pediatric Surgery
ant)20; submucosal Polidocanol (Aethoxysklerol: Base Pharma, Gordon, Australia; Sclerovein: sclerosant; Chemische Fabrik Kreussler & Co. GmbH, Wiesbaden, Germany) þ n-BCA20; bronchial brush þ fibrin glue8; holmium laser þ fibrin glue21; electrocautery þ fibrin glue22; submucosal polidocanol þ n-BCA23; mechanical abrasion or electrocautery þ n-BCA and lipiodol24; silver nitrate bead þ “glue”25; diarthermy þ fibrin glue13; bronchial brush þ fibrin glue26; endoscopic brush þ biosynthetic mesh (Surgisis: Cook Inc., Bloomington, Indiana, United States)27; diarthermy þ fibrin glue and aprotinin28; bronchial brush þ n-BCA and lipiodol10; biopsy forceps þ submucosal dextranomer/hyaluronic acid copolymer (Deflux: Oceana Therapeutics, Dublin, Ireland)29; endoscopic brush þ porcine dermal biological mesh (Permacol: TissueScience, Hampshire, United Kingdom) held in place by a synthetic absorbable sealant Duraseal (Confluent Surgical Inc., Waltham, Massachusetts, United States)(polyethylene glycol þ trilysine amine).30
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It is recommended at least 5 weeks after the initial repair when the residual inflammatory reaction in the area has subsided. The child can be placed on nasogastric (NG) feeds, and this also allows for respiratory symptoms to settle before surgery.4 The fistula should be cannulated (Fig. 1) at bronchoscopy to enable its identification during dissection.31–33 Depending on the level of the fistula an anterior neck crease or right thoracotomy is used to safely ligate the fistula, and suture the ends of the esophagus and trachea.4,34–37 Some have used a left extrapleural approach,38 a transcervical transtracheal approach,39 or via a median sternotomy.40 Many authors recommend placement of an interposition tissue between the sutured ends of the esophagus and trachea to prevent a second recurrence; these include mediastinal pleura,41–45 vascularized pedicle of pericardium,31,32,46 pedicled sternocleidomastoid flap,40 coastal cartilage graft,47 and omental flaps.48 The postoperative care is similar to after an ET.
Summary of Published Reports The outcomes after these two repairs are summarized in ►Tables 1–4. Overall, of the 57 patients managed using a variety of ETs there was an 84% (n ¼ 48) success rate compared with 93.5% (n ¼ 101) of 108 patients that had OS. Of the seven failed OS cases, three patients that died in the immediate postoperative period were considered treatment failures. One patient went on to have a cervical esophagostomy followed by gastric transposition,31 another two underwent an esophagectomy and gastric transposition each,32 and one
This document was downloaded for personal use only. Unauthorized distribution is strictly prohibited.
366
3
1
Bhatnagar et al (1999)
Hoelzer et al (1999)c
European Journal of Pediatric Surgery
Khuranna et al (2004)
6
1
2
Wiseman (1995)
Lopes et al (2003)
1
Gutierrez et al (1994)
1
1
Vandenplas et al (1993)
McGahren et al (2001)
1
Al-Samarrai et al (1987)
1
2
Rangecroft et al (1984)
NG et al (1999)
n
References (23 articles)
13.6 (1–54)
11
3
–
72
2.6 (1–5)
25.5 (6–45)
0.5
Vol. 24
2.2 (1–3)
1
3
1
1
2.5 (1–4) (four attempts using laser)
2.5 (1–4)
1
4
1
5.5 (5–6)
Mean number of treatments required (range)a
1 (16%)
0
0
0
0
1 (33%) using electrocautery
Ongoing treatment in Px 1
0
0
0
0
Treatment failures converted to open surgery n (%)
53 (16 -96)
36
42
24
48
3
6 (Px 2 only)
3
12
24
12
Mean length of follow-up in mo (range)
Diathermy coagulation
n-BCA via bronchoscopy þ submucosal polidocanol via esophagoscopy.
De-epithelialization with cautery þ application of fibrin glue.
De-epithelialization by coagulation of fistula epithelium with holmium laser þ fibrin glue sealant.
Fibrin glue
(Continued)
Unspecified number of previous failed open repairs.
Three previous open repairs and one endoscopic gluing with fibrin failed.
Two previous failed endoscopic attempts with fibrin glue.
AL
Three AL
Electrocautery ¼ 2 Nd:YAG laser ¼ 1
AS; one previous failed open repair.
Two previous failed open repairs.
One previous failed open repair.
Notes
One previous failed endoscopic approach with electrocautery and n-BCA followed by failed thoracotomy with pleural flap. One AS. Respiratory distress in two using electrocautery
Postoperative complications
De-epithelialization with bronchial brush þ injection with fibrin glue.
Fibrin glue (Tisseel, Tissucol, Tisseal).
Fibrin glue; n-BCA; n-BCA þ submucosal 30% NaCl; n-BCA þ submucosal Polidocanol (Aethoxysklerol).
n-butyl cyanoacrylate (n-BCA, enbucrilate, Histoacryl) þ submucosal injection of 0.5% Polidocanol (Aethoxysklerol, Sclerovein) via esophagoscopy.
De-epithelialization (fulguration of fistula mucosa with diathermy).
Endoscopic technique (sealant þ/ de-epithelializationb)
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1 (16%)
1 (100%)
0
1 (100%)
1 (100%)
1 (33%) (using electrocautery)
1 (50%)
1 (100%)
0
1 (100%)
–
12
0
Successful single treatments n (%)
60 (1 Px)
Mean age at repair in mo (range)
Table 1 Summary of published endoscopic treatments of recurrent tracheoesophageal fistulas
Management of RTEF: A Systematic Review Aworanti, Awadalla
No. 5/2014
367
European Journal of Pediatric Surgery
Vol. 24
3
1
4
3
1
1
1
Meier et al (2007)c
Keckler et al (2008)
Richter et al (2008) c
Sung et al (2008)
Yoon et al (2009)
Farra et al (2010)
Rakoczy et al (2010)
4
Linder (2006)
7
6
Tzifa et al (2005)
San Romàn et al (2006)
n
References (23 articles)
Table 1 (Continued)
No. 5/2014
1 (100%)
– 1
1
1
3.3 (3–4)
1.2 (1–2)
3
2 (1–3)
1.5 (1–3)
4.3 (4–5)
1.6 (1–2)
Mean number of treatments required (range)a
0
0
0
0
0
0
1 (33%)
1 (14%)
1 (25%)
0
Treatment failures converted to open surgery n (%)
12
22
8
14 (9–22)
16
36
48 (?–60)d
89 (24–132)
45 (24–72)
66 (36–108)
Mean length of follow-up in mo (range)
Potassium titanyl phosphate laser
Submucosal injection of fibrin glue.
De-epithelialization with brush þ application of n-BCA þ Lipiodol glue via esophagus.
Chemocauterization using 50% trichloroacetic acid.
De-epithelialization with a Bugbee fulgurating diathermy electrode then sealed with fibrin glue þ added aprotinin.
Two previous failed endoscopic attempts with electrocautery þ fibrin glue.
AS
AS. Three previous failed open repairs.
One AS. Two previous failed open repairs.
One previous failed open repair with pleural interposition.
De-epithelialization þ plugging with a biosynthetic mesh (Surgisis) and fibrin glue at first attempt only
Notes
One previous failed open repair using a pleural interposition flap. Respiratory distress
One mortality case 3 days postoperatively. Cause bugbee electrocautery.
Postoperative complications
De-epithelialization with bronchial brush þ fibrin glue.
De-epithelialization by coagulation of fistula mucosa with diathermy in 4 Px þ fibrin glue sealant in all.
De-epithelialization with silver nitrate bead þ “ glue” and use of esophageal balloon to occlude airflow through fistula.
De-epithelialization by mechanical abrasion or bug by electrocautery followed by injection of n-BCA þ lipiodol into the fistula.
Endoscopic technique (sealant þ/ de-epithelialization b)
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1 (100%)
1 (100%)
0
3 (75%)
0
0.5
156
58 (3–156)
11.5 (3–20)
12
1 (33%)
3 (43%)
0
2 (33%)
Successful single treatments n (%)
Management of RTEF: A Systematic Review
39 (5–108)
0.55 (0.46–0.66)
18.9 (12–156)
16.5 (2–60)
Mean age at repair in mo (range)
368 Aworanti, Awadalla
5
1
57
Briganti et al (2011)
van Niekerk (2012)
Total/range
1 (100%)
21 (37%)
0.5–156e
0
Successful single treatments n (%)
6
7.8 (1–18)
Mean age at repair in mo (range)
2.2 (1–6) 106 procedures in 49 successful cases including 1 ongoing.
1
2
Mean number of treatments required (range)a
9 (16%)
0
3 (60%)
Treatment failures converted to open surgery n (%)
3–108e
De-epithelialization with endoscopic brush þ plugging with a porcine dermal biological mesh (Permacol) held in place by a synthetic absorbable sealant. Duraseal (polyethylene glycol þ trilysine amine)
De-epithelialization with biopsy forceps þ dextranomer/hyaluronic acid copolymer (Deflux) injected submucosaly.
–
24
Endoscopic technique (sealant þ/ de-epithelialization b)
Mean length of follow-up in mo (range)
Three (5%) respiratory distress; 1 (1.7%) mortality
Postoperative complications
Four (7%) previous leaks (AL); 10 (17%) previous strictures (AS); 16 previous failed open repairs; 6 previous failed alternative endoscopic Tx.
AS in all
Notes
European Journal of Pediatric Surgery
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Abbreviations: AL, anastomotic leaks before RTEF; AS, associated stricture before RTEF; mo, months; n, number; Px, patient; RTEF, recurrent tracheoesophageal fistula; Tx, treatment. Note: n-Butyl cyanoacrylate (n-BCA; Enbucrilate, Histoacryl); fibrin glue (Tisseel, Tissucol, Tisseal). a Mean number of treatments required for successfully treated cases. b De-epithelialization involves abrading, denuding, cauterizing, chemocauterizing, sclerosing, or destroying the epithelial lining of the fistula using bronchial brush, biopsy forceps, bead of silver nitrate, fulgurating diarthermy, electrocautery, potassium titanyl phosphate (KTP) laser, trichloroacetic acid (TCA), polidocanol (sclerosant), Nd:YAG laser, or holmium laser. c Literature review. d Median. e Reflects the range of available and compatible data only.
n
References (23 articles)
Table 1 (Continued)
Management of RTEF: A Systematic Review Aworanti, Awadalla
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369
European Journal of Pediatric Surgery
4
Girdany et al (1956)
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No. 5/2014
3 (100%) 3 (100%)
5.6 (2–11)
–
3
3
2
22
1
1
18
1
1
1
1
Kafrouni et al (1970) b
Stanford et al (1973)
Slim et al (1974)
Ein et al (1983)
Botham et al (1986)
Martin et al (1986)
Ghandour et al (1990)
Narasimhan et al (1990)
Vos et al (1996)
Holland et al (1998)
Delarue et al (2002)
0.5
11
90
0
2
1
1
1
1.2 (1–2)
1
1
1.2 (1–2)
1
0
0
0
0
1 (esophageal replacement)
0
0
0
0
0
0
1 (mortality)
1 (mortality)
0
1 (mortality)
0
Treatment failures converted to other procedure n (%)
27
4
Previous tracheal injury and AL. Subsequent planned esophageal reconstruction with colonic interposition.
Coastal cartilage graft þ fibrin sealant
AS
Nine AL
One previous failed open repair.
AL. One previous failed repair with pleural flap.
(9 of 23 Px) 39% AL; (10 of 23 Px) 43% AS.
Two AS and two AL. One Px also had stricture resection.
One AS
Three AS. One Px also had stricture resection.
Two AS
AS
Notes
Previous failed single open and multiple endoscopic attempts at repair.
Five leaks, 1 death; 10 mo postoperatively
Leak at the fistula site in four
One mortality
Esophageal leak þ esophagopleurocutaneous fistula. 1 mortality (patient died)
One mortality
Complications
Pedicled sternocleidomastoid flap via median sternotomy.
Pleural flap
Mediastinal pleura
– 144
Unspecified
Transcervical transtracheal repair
– –
Vascularized pedicle of pericardium
Simple division of fistula and suturing of ends
8
–
Left extra pleural approach
–
–
– Pleural flap
Closure with fine 6–0 monofilament stainless steel wire sutures.
–
–
“Tip of lung” interposed
0.5
Mediastinal pleura
Unspecified
– 12
Pleural flap
Surgical technique/ interposition tissue
5
Mean length of follow-up in mo (range)
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1 (100%)
1
1
14 (78%)
–
12
1
10
1
17 (77%)
–
5
2 (100%)
1
1
1
1
1
1.7 (1–2)
1
Mean number of treatments required (range)a
Management of RTEF: A Systematic Review
10.5 (5–16)
2 (66%)
5.3 (1–14)
3
Cowley (1967)
0
1 (100%)
1 (25%)
1 (100%)
Successful single treatments n (%)
1
3
23
5.75 (1–16)
24
Mean age at repair in mo (range)
Falletta (1964)
1
1
Hewlett (1955)
Moskovitz et al (1960)
n
References (21 articles)
Table 2 Summary of published open repairs of recurrent tracheoesophageal fistulas
370 Aworanti, Awadalla
1
2
4
26
11
108
Hotta et al (2002)
Bonnard et al (2007)
Briganti et al (2009)
Bruch et al (2010)
Coran (2013)
Total/range
4 (100%)
23 (88%)
6 (54%)
85 (79%)
–
–
0.5–90 d
2 (100%)
1
Successful single treatments n (%)
23.7 (12–30)
2.7 (1.7–3.6)
4
Mean age at repair in mo (range)
1.1 (1–2) 117 procedures in 101 successful cases
1.3 (1–2)
1.1 (1–2)
1
1
1
Mean number of treatments required (range) a
7 (6.5%; 3 early postoperative mortalities)
2 (both had esophagectomy and gastric transposition)
1 (cervical esophagostomy followed by gastric pull-up)
0
0
0
Treatment failures converted to other procedure n (%)
0.5–287d
–
–
17 (16%) leaks; 4 (3.7%) mortality
–
European Journal of Pediatric Surgery
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40 (37%) previous leaks (AL); 25 (23%) previous strictures (AS); 4 previous failed alternative open repair; 9 previous failed endoscopic Tx.
–
18 (68%) AS. Three had previous failed endoscopic Tx.
Seven leaks, four strictures.
84 (6–287)c Pleura n ¼ 11 Pericardium n ¼ 8 Muscle flap n ¼ 3 Lymph nodes n ¼ 2 No tissue n ¼ 1
–
–
One previous failed repair each. One endoscopic gluing and the other open using a pleural interposition flap. One AS.
Notes
Complications
Four AS. Three previous failed endoscopic Tx.
Omental flap interposition
Ligation and division of fistula
Surgical technique/ interposition tissue
(18–60)
20.5 (17– 24)
6
Mean length of follow-up in mo (range)
Abbreviations: AL, anastomotic leaks before RTEF; AS, associated stricture before RTEF; n, number; Px, patient; RTEF, recurrent tracheoesophageal fistula; Tx, treatment. Note: n-Butyl cyanoacrylate (n-BCA; Enbucrilate; Histoacryl); fibrin glue (Tisseel, Tissucol, Tisseal). a Mean number of treatments required for successfully treated cases. b Literature review. c Median. d Reflects the range of available and compatible data only.
n
References (21 articles)
Table 2 (Continued)
Management of RTEF: A Systematic Review Aworanti, Awadalla
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371
Management of RTEF: A Systematic Review
Aworanti, Awadalla
Table 3 Summary of published outcomes of endoscopic treatment versus open surgery for recurrent tracheoesophageal fistulas Descriptive data
Endoscopic treatment
Open surgery
Number of patients
57
108
Age range at treatment onset in moa
0.5–156
0.5–90
Successful single treatments n (%)
21 (37%)
85 (79%)
Mean number of treatments required (range)
2.1 (1–6)
1.1 (1–2)
Treatment failures converted to other procedure n (%)
9 (16%) (84% overall success)
7 (6.5%) (93.5% overall success including three perioperative mortalities)
Mean length of follow-up in mo (range)a
3–108
0.5–287
Abbreviation: mo, months. a Range for available data only.
Table 4 Summary of results comparing de-epithelialization only versus sealant only versus de-epithelialization þ sealant Descriptive data
De-epithelialization only (5 articles)
Sealant only (4 articles)
De-epithelialization þ sealant (15 articles)
Number of patients
15
6
36
Successful single treatments n (%)
3 (20%)
4 (67%)
14 (39%)
Mean number of treatments performed (range)
2.7 (1–6)
1.5 (1–3)
2 (1–5)
Treatment failures converted to other procedure n (%)
2 (13%)
1 (16.6%)
6 (16.6%)
Note: When multiple techniques were used on one patient the last technique described was used to deduce the final outcome.
had an unspecified esophageal replacement.37 The refistulation rate after OS was 21% and an average of 1.1 (range 1–2) procedures were required to achieve treatment success. However after ET, treatment was either incomplete or resulted in refistulation in 63%, and though only an average of 2.1 treatments were required to achieve successful closure, occasionally up to 6 treatments were necessary.15 Both averages excluded several treatment attempts in unsuccessful cases that were subsequently converted to an alternative treatment modality; the failed ET cases were successfully treated by OS. These results were reported after a maximum follow-up of 9 years and 23 years for ET and OS, respectively. The major complications after OS were 17 (16%) leaks and 4 (3.7%) deaths; 1 of these deaths occurred 10 months after surgery and a second RTEF was found at autopsy.37 These mortalities after OS were reported in 1956, 1964, 1967, and 1990. After ET, three suffered significant respiratory distress postoperatively and there was 1 (1.7%) death reported in 2005; this occurred 3 days postprocedure and the cause was unspecified.24 The results when ET were grouped into “de-epithelialization only,” “sealant only,” and “de-epithelialization combined with sealants” (as suggested by Richter et al28) is summarized in ►Table 4. There were no significant observations except that contrary to Richter’s findings, the use of “sealants only” resulted in the most successful single treatments reported (67%) and the least number of treatments required (mean 1.5; range 1–3). This finding should however be interpreted with caution due to the small numbers, particularly in the “sealant only” group (n ¼ 6). European Journal of Pediatric Surgery
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Discussion Apart from inadequate ligation in the first instance, these fistulas probably recur due to an anastomotic leak that forms a localized abscess which bores into the trachea at the point of least resistance: the suture lines. Strictures that require forceful dilatations can lead to the same phenomenon.26,32,34,36,46 This review suggest that one in four and one in five cases of RTEF reported in the literature document an association with leaks or strictures, respectively. The mean time to recurrence is 2 months,4,31 but have been reported up to 32 years after the initial repair.49 The usual clinical presentation includes persistent coughing due to saliva aspiration, choking, and apnea during feeds, recurrent aspiration
Fig. 1 An operative view via an anterior neck dissection showing easy access to the trachea (a), the cannulated fistula (arrow), and the retracted esophagus (b).
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372
pneumonia, and eventually chronic lung disease.4,24,28,37 A prone esophagram performed by injecting the contrast under pressure via an NG tube while withdrawing the tube is the most sensitive investigation with the least false negatives.4,35,50 The diagnosis is confirmed at bronchoscopy or esophagoscopy occasionally requiring the aid of methylene blue.7,26,31,32 Important differentials are tracheal diverticula51 or a missed second fistula.52,53 When a RTEF has being diagnosed, OS was the only management option available until 1974 when ET was first described by Gdanietz et al.54 Since then there has been a variety of endoscopic techniques with varying results published. The drive to develop and improve these new techniques stem from the belief that open redo surgery is technically challenging and plagued with a high morbidity, mortality, and further recurrences. Most authors that quote these poor expectations directly or indirectly reference Kafrouni et al review from19707 where they reported a 59% mortality after RTEF.10–14,20,24,26,28,55 However, on further analysis of their review, the actual mortality after redo surgery for RTEF was 32% (9 of 28 reoperated cases). Manning et al as early as 1986 has reported a fall in operative mortality rates after primary TEF repair from 56 to 6.9%, since Haight’s pioneering repair performed at their center5; one would then expect that the mortality after surgery for RTEF should show a similar improvement. Our review of the published reports of OS for RTEF from 1955 to 2013 revealed only three perioperative deaths that all occurred in the 60s. The major complications after OS were leaks and second recurrences; another redo surgery for a second recurrence will be challenging for obvious reasons. In this review, a second recurrence (after OS) was reported in 21% of the reported RTEF cases, comparable to 22, 22, and 11.5% reported in the OS patient series by Ein et al (1983),4 Ghandour et al (1990),37 and Bruch et al (2010),31 respectively. The use of a vascularized interposition tissue is recommended to prevent a second RTEF. No more than two OS treatments are usually required and treatment failures resulted in some form of esophageal replacement reported in only four patients. Hence, this updated review shows that OS can be safely performed without a high morbidity and mortality. Minimally invasive techniques are widely accepted to be better tolerated by patients and whenever possible such options should be explored, thus supporting the case for ET; however, the outcomes should be comparable. Several authors have used a variety of techniques for ET, summarized above, all hoping to achieve better outcomes. There have been three literature reviews on ET. In Hoelzer 1999 review,12 15 cases were reported, having been treated with diarthermy, laser, n-BCA, and fibrin glue, and 1 to 5 treatments were necessary with complete closure in only 10 patients (67%). Meier et al in 200726 reported an overall success rate of 60% of 62 patients culled from the literature (fibrin glue 55% [n ¼ 22], n-BCA 48% [n ¼ 29], laser 80% [n ¼ 5], diathermy 50% [n ¼ 10], n-BCA þ sclerosant100% [n ¼ 5]); the mean number of applications ranged from 1 to 5.5. They concluded that ET is safe, effective, has less morbidity than OS and recommended fibrin glue but advised close follow-up. Richter et al28 in their 2008 review, culled 37 patients from the literature and categorized ET into: application of sealant only
Aworanti, Awadalla
(n ¼ 14; 78.6% success), de-epitheliazation of the fistula tract only (n ¼ 8; 62.5% success), de-epitheliazation combined with sealant (n ¼ 15; 93.3%). Overall there was an 81.1% success rate (48.6% after a single attempt) and they concluded that deepitheliazation with diathermy combined with fibrin glue sealant will yield the best results; fibrin glue by nature would work better if the opposing surfaces are closely apposed and not epithelialized.21 The failures seen with ET may be partly because the sealants fibrin glue and n-BCA are biodegradable. Aprotinin, an antifibrinolytic, prolongs the stability of fibrin glue in vivo for up to 10 days only56; however, the fibrin clot formed has a loose scaffold that encourages in-growth of fibroblasts. n-BCA, commonly used for skin closure, is not recommended for mucosal application by most manufacturers, as its stability and possible absorption in such environments is not yet fully determined. Willets et al (1998)55 also appreciating the possibility of late refistulation after ET, and the limited follow-up period of the reported cases managed endoscopically, decided to evaluate the long-term outcome of these patients by contacting the respective authors. They concluded that of 22 patients initially reported to have had successful ET, only 12 (55%) remained closed after a median follow-up of 107 months; there was however no morbidity or mortality directly related to these procedures. These findings somewhat mirror ours (84% overall success after a range of 1–6 ET). However, there were three reports of significant respiratory distress following ET; aspiration of the sealant or perforation of the tracheal wall when using diathermy or laser for de-epitheliazation are serious potential hazards.8,23,29 Furthermore, these patients that require up to six ETs suggest that they are representing with the typical symptoms of saliva and feed aspiration that, aside from the burden to the family of frequent hospitalization, can eventually lead to chronic lung disease. Unless there is a systematic way of evaluating for refistulation after ET, lung morbidity may be unavoidable. On the other hand, no child required more than two open operations for RTEF, and anastomotic leaks, usually managed conservatively, were the major complications to contend with. The main limitations of this review include the heterogeneity of the included articles, particularly as case reports would predominantly report positive outcomes. Furthermore, some authors occasionally mentioned a variety of previous failed OS and/or ET attempts in the patients whom they formally published their definitive repair on; these previous repairs (due to insufficient data on them) could not be considered when analyzing our results. However, they are mentioned in ►Tables 1 and 2.
Conclusion In conclusion, based on our findings on 165 published cases, OS for the repair of recurrent TEF has a low morbidity, mortality, and re-fistulation rate particularly when an interposition tissue is placed between the trachea and esophagus. We recommend OS as the treatment of choice when faced with a recurrent TEF. Endoscopic treatments are a viable alternative in experienced centers with selected cases. Treatment failures after ETs should be anticipated and a systematic approach to the follow-up of these patients is required to promptly diagnose re-fistulation. European Journal of Pediatric Surgery
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Management of RTEF: A Systematic Review
Management of RTEF: A Systematic Review
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Further clinical trials and controlled experiments are necessary to devise the technique and ideal, preferably nonbiodegradable, sealant that guarantees a definitive endoscopic closure of a recurrent TEF after one attempt.
19
20 21
Conflict of Interest None.
22
References
23
1 Haight C, Towsley HA. Congenital atresia of the esophagus with
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7 8 9
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tracheoesophageal fistula: extrapleural ligation of fistula and endto-end anastomosis of esophageal segments. Surg Gynecol Obstet 1943;76:672–688 Holcomb GW III, Rothenberg SS, Bax KM, et al. Thoracoscopic repair of esophageal atresia and tracheoesophageal fistula: a multi-institutional analysis. Ann Surg 2005;242(3):422–428, discussion 428–430 Aziz GA, Schier F. Thoracoscopic ligation of a tracheoesophageal Htype fistula in a newborn. J Pediatr Surg 2005;40(6):e35–e36 Ein SH, Stringer DA, Stephens CA, Shandling B, Simpson J, Filler RM. Recurrent tracheoesophageal fistulas seventeen-year review. J Pediatr Surg 1983;18(4):436–441 Manning PB, Morgan RA, Coran AG, et al. Fifty years’ experience with esophageal atresia and tracheoesophageal fistula. Beginning with Cameron Haight’s first operation in 1935. Ann Surg 1986; 204(4):446–453 Myers NA, Beasley SW, Auldist AW. Secondary esophageal surgery following repair of esophageal atresia with distal tracheoesophageal fistula. J Pediatr Surg 1990;25(7):773–777 Kafrouni G, Baick CH, Woolley MM. Recurrent tracheoesophageal fistula: a diagnostic problem. Surgery 1970;68(5):889–894 Wiseman NE. Endoscopic closure of recurrent tracheoesophageal fistula using Tisseel. J Pediatr Surg 1995;30(8):1236–1237 Al-Samarrai AY, Jessen K, Haque K. Endoscopic obliteration of a recurrent tracheoesophageal fistula. J Pediatr Surg 1987;22(11): 993 Yoon JH, Lee HL, Lee OY, et al. Endoscopic treatment of recurrent congenital tracheoesophageal fistula with Histoacryl glue via the esophagus. Gastrointest Endosc 2009;69(7):1394–1396 Gutierrez C, Barrios JE, Lluna J, et al. Recurrent tracheoesophageal fistula treated with fibrin glue. J Pediatr Surg 1994;29(12): 1567–1569 Hoelzer DJ, Luft JD. Successful long-term endoscopic closure of a recurrent tracheoesophageal fistula with fibrin glue in a child. Int J Pediatr Otorhinolaryngol 1999;48(3):259–263 Gutiérrez San Román C, Barrios JE, Lluna J, et al. Long-term assessment of the treatment of recurrent tracheoesophageal fistula with fibrin glue associated with diathermy. J Pediatr Surg 2006;41(11):1870–1873 Farra J, Zhuge Y, Neville HL, Thompson WR, Sola JE. Submucosal fibrin glue injection for closure of recurrent tracheoesophageal fistula. Pediatr Surg Int 2010;26(2):237–240 Rangecroft L, Bush GH, Lister J, Irving IM. Endoscopic diathermy obliteration of recurrent tracheoesophageal fistulae. J Pediatr Surg 1984;19(1):41–43 Khurana S, Andrew Ford WD. Long-term results of endoscopic diathermy coagulation of recurrent tracheoesophageal fistula. Pediatr Endosurg Inno Tech 2004;8(1):31–34 Bhatnagar V, Lal R, Sriniwas M, Agarwala S, Mitra DK. Endoscopic treatment of tracheoesophageal fistula using electrocautery and the Nd:YAG laser. J Pediatr Surg 1999;34(3):464–467 Sung MW, Chang H, Hah JH, Kim KH. Endoscopic management of recurrent tracheoesophageal fistula with trichloroacetic acid che-
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mocauterization: a preliminary report. J Pediatr Surg 2008;43(11): 2124–2127 Rakoczy G, Brown B, Barman D, et al. KTP laser: an important tool in refractory recurrent tracheo-esophageal fistula in children. Int J Pediatr Otorhinolaryngol 2010;74(3):326–327 Vandenplas Y, Helven R, Derop H, et al. Endoscopic obliteration of recurrent tracheoesophageal fistula. Dig Dis Sci 1993;38(2):374–377 Ng WT, Luk HT, Lau CW. Endoscopic treatment of recurrent tracheo-oesophageal fistulae: the optimal technique. Pediatr Surg Int 1999;15(5–6):449–450 MacGahren ED, Rodgers BM. Bronchoscopic obliteration of recurrent tracheoesophageal fistula in an infant. Pediatr Endosurg Inno Tech 2001;5(1):37–42 Lopes MF, Pires J, Nogueria Brandão A, Reis A, Morais Leitão L. Endoscopic obliteration of a recurrent tracheoesophageal fistula with enbucrilate and polidocanol in a child. Surg Endosc 2003; 17(4):657 Tzifa KT, Maxwell EL, Chait P, et al. Endoscopic treatment of congenital H-Type and recurrent tracheoesophageal fistula with electrocautery and histoacryl glue. Int J Pediatr Otorhinolaryngol 2006;70(5):925–930 Linder A. Endoscopic sealing of tracheoesophageal fistulae. An improved method. J Bronchology 2006;13(4):188–190 Meier JD, Sulman CG, Almond PS, Holinger LD. Endoscopic management of recurrent congenital tracheoesophageal fistula: a review of techniques and results. Int J Pediatr Otorhinolaryngol 2007;71(5):691–697 Keckler SJ, St Peter SD, Calkins CM, Holcomb GW III. Occlusion of a recurrent tracheoesophageal fistula with surgisis. J Laparoendosc Adv Surg Tech A 2008;18(3):465–468 Richter GT, Ryckman F, Brown RL, Rutter MJ. Endoscopic management of recurrent tracheoesophageal fistula. J Pediatr Surg 2008; 43(1):238–245 Briganti V, Coletta R, Giannino G, Calisti A. Usefulness of dextranomer/hyaluronic acid copolymer in bronchoscopic treatment of recurrent tracheoesophageal fistula in children. Int J Pediatr Otorhinolaryngol 2011;75(9):1191–1194 van Niekerk ML. The treatment of recurrent tracheo-oesophageal fistula with biosynthetic mesh. S Afr J Surg 2012;50(3):106 Bruch SW, Hirschl RB, Coran AG. The diagnosis and management of recurrent tracheoesophageal fistulas. J Pediatr Surg 2010;45(2): 337–340 Coran AG. Redo esophageal surgery: the diagnosis and management of recurrent tracheoesophageal fistula. Pediatr Surg Int 2013;29(10):995–999 Hotta Y, Uezono S, Segawa O, et al. Precise localization of a recurrent tracheo-oesophageal fistula using retrograde guide wire placement. Paediatr Anaesth 2002;12(6):541–543 Girdany BR, Sieber WK. Tracheoesophageal fistula without esophageal atresia—congenital and recurrent. Pediatrics 1956;18(6):935–942 Falletta GP. Recommunication on repair of congenital tracheoesophageal fistula. Report of a case and review of literature. Arch Surg 1964;88:779–786 Cowley LL. Congenital tracheo-esophageal fistula: recurrence after repair. Am Surg 1967;33(5):409–410 Ghandour KE, Spitz L, Brereton RJ, Kiely EM. Recurrent tracheooesophageal fistula: experience with 24 patients. J Paediatr Child Health 1990;26(2):89–91 Slim MS, Tabry IF. Left extrapleural approach for the repair of recurrent tracheoesophageal fistula. J Thorac Cardiovasc Surg 1974;68(4):654–657 Martin LW, Cox JA, Cotton R, Oldham KT. Transtracheal repair of recurrent tracheoesophageal fistula. J Pediatr Surg 1986;21(5): 402–403 Holland AJ, Ford WD, Guerin RL. Median sternotomy and use of a pedicled sternocleidomastoid muscle flap in the management of recurrent tracheoesophageal fistula. J Pediatr Surg 1998;33(4): 657–659
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J Thorac Surg 1955;29(6):658–664 Moskovitz WS, Hughes CW, Bowers WF. Recurrent tracheoesophageal fistula. Am J Surg 1960;100:110–112 Stanford W, Armstrong RG, Cline RE, Williams MJ. Recurrent tracheoesophageal fistula. Ann Thorac Surg 1973;15(5):452–455 Narasimhan KL, Rao KL, Mitra SK. Management of recurrent tracheoesophageal fistula. Indian Pediatr 1990;27(10):1114–1116 Vos A, Ekkelkamp S. Congenital tracheoesophageal fistula: preventing recurrence. J Pediatr Surg 1996;31(7):936–938 Botham MJ, Coran AG. The use of pericardium for the management of recurrent tracheoesophageal fistula. J Pediatr Surg 1986;21(2): 164–166 Delarue A, Paut O, Simeoni J, Lepra SB, Nicollas R. Costal cartilage grafting for repair of a recurrent tracheoesophageal fistula in a 1.6-kg baby with esophageal atresia. Pediatr Surg Int 2002;18(2-3):162–164 Bonnard A, Paye-Jaouen A, Aizenfisz S, El Ghoneimi A. Laparoscopically harvested omental flap for recurrent tracheoesophageal fistula in a newborn baby. J Thorac Cardiovasc Surg 2007;134(6): 1592–1593 Ashour M. Recurrence of tracheo-oesophageal fistula 32 years after primary repair. Thorax 1986;41(1):76–77
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protocol for investigation. Radiology 1984;151(3):637–641 51 Shah AR, Lazar EL, Atlas AB. Tracheal diverticula after tracheoe-
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sophageal fistula repair: case series and review of the literature. J Pediatr Surg 2009;44(11):2107–2111 Guo W, Li Y, Jiao A, Peng Y, Hou D, Chen Y. Tracheoesophageal fistula after primary repair of type C esophageal atresia in the neonatal period: recurrent or missed second congenital fistula. J Pediatr Surg 2010;45(12):2351–2355 Schulte T, Ankermann T, Claas A, Engler S. An extremely rare abnormality of a double tracheoesophageal fistula without atresia of the esophagus; a case report and review of the literature. J Pediatr Surg 2009;44(10):e9–e12 Gdanietz K, Wiesner B, Krause I, Mau H, Jung FJ. Tissue-adhesive for sealing of oesophagotracheal fistulae in children (author’s transl) [in German]. Z Erkr Atmungsorgane 1974; 141(1):46–50 Willetts IE, Dudley NE, Tam PK. Endoscopic treatment of recurrent tracheo-oesophageal fistulae: long-term results. Pediatr Surg Int 1998;13(4):256–258 Seelich T. Tissucol (Immuno, Vienna): biochemistry and methods of application. Head Neck Pathol 1982;3:65–69
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41 Hewlett TH. Recurrent tracheo-esophageal fistula; case report.
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Management of Recurrent Tracheoesophageal Fistulas: A Systematic Review Olugbenga Aworanti1,2
Sami Awadalla1
1 Department of Paediatric Surgery, Children’s University Hospital,
Dublin, Ireland 2 Department of Paediatric Surgery, Our Lady’s Children’s Hospital, Crumlin, Dublin, Ireland
Address for correspondence Olugbenga Aworanti, MBBS, MRCSEd, MSc, Department of Paediatric Surgery, Children’s University Hospital, Temple Street, Dublin 1, Ireland (e-mail: [email protected]).
Abstract
Keywords
► recurrent tracheoesophageal fistula ► systematic review ► endoscopic ► management
Introduction The management of a recurrent tracheoesophageal fistula (RTEF) includes either open surgery (OS) or an endoscopic treatment (ET); the ideal option is unclear. We aim to comparatively review all published treatment options, and outcomes, for managing RTEF. Materials and Methods A literature search was performed using the keywords “recurrent tracheoesophageal fistula.” All English language articles describing the management of RTEF in children were reviewed. A synthesis of the relevant data is presented in a descriptive form due to the heterogeneity of the included articles. Results A total of 44 papers between 1955 and 2013 described 165 patients; 57 ET and 108 OS. Of the 57 ET patients, there was an 84% success rate compared with 93.5% of 108 OS patients; the failed ET cases were all successfully treated by OS. The refistulation rate after OS was 21% and an average of 1.1 (range 1–2) procedures were required. After ET, the refistulation rate was 63% and an average of 2.1 (range 1–6) treatments were required for success; these results were reported after a maximum follow-up of 9 years and 23 years for ET and OS, respectively. The major complications after OS were 17 (16%) leaks and 4 (3.7%) deaths, while for ET 3 (5%) suffered respiratory distress postoperatively and there was 1 (1.7%) death. Conclusion OS for RTEF has a low morbidity and mortality, a higher success rate, and requires fewer treatments than an endoscopic repair. The ideal ET is undecided but it remains a viable alternative provided treatment failures are anticipated and prompt redo treatments initiated to prevent ongoing respiratory morbidity.
Introduction The spectrum of esophageal atresia (EA) and tracheoesophageal fistula (TEF) include an isolated EA, EA with a proximal and/or distal TEF and the “H” or “N” type fistula. Since the initial description of the open surgical repair1 and subsequent thoracoscopic approaches2,3 the outcomes have been very encouraging. Anastomotic leaks, esophageal strictures, gastroesophageal reflux, and tracheomalacia are complications
received October 3, 2013 accepted after revision January 11, 2014 published online March 28, 2014
pediatric surgeons are familiar with. The incidence of a fistula recurrence range from 1.9 to 11% after open and thoracoscopic repairs.2,4–8 The diagnosis of these recurrent tracheoesophageal fistulas (RTEF) can often be elusive to the unsuspecting physician and the ideal approach to their management is still in debate. Proponents of an endoscopic approach to this problem frequently state that the morbidity, mortality, and second recurrence rate are too high with an open approach and majority directly or indirectly reference
© 2014 Georg Thieme Verlag KG Stuttgart · New York
DOI http://dx.doi.org/ 10.1055/s-0034-1370780. ISSN 0939-7248.
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Eur J Pediatr Surg 2014;24:365–375.
Management of RTEF: A Systematic Review
Aworanti, Awadalla
the only literature review of open surgery (OS) from 1970 by Kafrouni et al.7 Here, we reviewed the published treatment options, and their outcomes, for managing RTEF. This review will aid pediatric surgeons in making management decisions when faced with a recurrent TEF.
Patients/Materials and Methods A Medline, Embase, and web of science searches were performed using the keywords recurrent tracheoesophageal fistula. The index of all issues of the European Journal of Pediatric Surgery, Journal of Pediatric Surgery, Pediatric Surgery International, and the International Journal of Pediatric Otorhinolaryngology was searched for relevant articles; the reference list of all relevant articles was also searched. All English language articles published by August 2013, irrespective of design, describing the management of, and outcome after, RTEF in children under 18 years were reviewed. The data obtained included number of patients treated, treatment modality employed, effectiveness of each treatment modality, and the outcomes after treatment. These outcomes include the successful single treatments, number of treatments required, number of treatment failures requiring conversion to an alternate treatment modality, and the incidence of postoperative complications. A synthesis of the data is presented in a descriptive form due to the heterogeneity of the included articles.
Results
Some perform a repeat esophagoscopy and bronchoscopy to confirm closure at both ends of the fistula, exclude dislodgement/aspiration of remnant sealant, and exclude intraluminal damage particularly when electrocautery or laser de-epithelialization is performed. The child is usually kept nil per oral for several days and a follow-up contrast esophagram is obtained before oral feeding.
Open Surgical Repair
A total of 23 papers between 1984 and 2012 described 57 patients undergoing some form of endoscopic treatment (ET) while 21 papers between 1955 and 2013 described 108 patients that underwent an open surgical repair (►Tables 1 and 2). A total of 165 cases of RTEF are described and there were 44 (26.7%) cases associated with anastomotic leaks while 35 (21.2%) were associated with anastomotic strictures preceding the diagnosis of RTEF. Below is a summary of the published operative principles.
Endoscopic Treatment This is usually performed via bronchoscopy but can be done via esophagoscopy.9,10 Three categories of ET have been described as follows: • Injection of a sealant into the fistula tract or submucosa. The sealants commonly used are fibrin glue (Tisseel: Baxter Corporation, Ontario, Canada; Tissucol: Baxter, Vienna, Austria; and Tisseal: Baxter Inc., Deerfield, Illinois, United States)11–14 and n-butyl cyanoacrylate (n-BCA, enbucrilate, and Histoacryl: B. Braun Melsungen AG, Mesungen, Germany). • De-epithelialization of the fistula tract involves abrading, denuding, cauterizing, fulgurating, chemocauterizing, sclerosing, or destroying the epithelial lining of the fistula using diarthermy,15,16 Nd:YAG laser,17 electrocautery,17 trichloroacetic acid,18 or potassium titanyl phosphate laser.19 • A combination of de-epithelialization and injection of a sealant: submucosal 30% NaCl (sclerosant) þ n-BCA (sealEuropean Journal of Pediatric Surgery
ant)20; submucosal Polidocanol (Aethoxysklerol: Base Pharma, Gordon, Australia; Sclerovein: sclerosant; Chemische Fabrik Kreussler & Co. GmbH, Wiesbaden, Germany) þ n-BCA20; bronchial brush þ fibrin glue8; holmium laser þ fibrin glue21; electrocautery þ fibrin glue22; submucosal polidocanol þ n-BCA23; mechanical abrasion or electrocautery þ n-BCA and lipiodol24; silver nitrate bead þ “glue”25; diarthermy þ fibrin glue13; bronchial brush þ fibrin glue26; endoscopic brush þ biosynthetic mesh (Surgisis: Cook Inc., Bloomington, Indiana, United States)27; diarthermy þ fibrin glue and aprotinin28; bronchial brush þ n-BCA and lipiodol10; biopsy forceps þ submucosal dextranomer/hyaluronic acid copolymer (Deflux: Oceana Therapeutics, Dublin, Ireland)29; endoscopic brush þ porcine dermal biological mesh (Permacol: TissueScience, Hampshire, United Kingdom) held in place by a synthetic absorbable sealant Duraseal (Confluent Surgical Inc., Waltham, Massachusetts, United States)(polyethylene glycol þ trilysine amine).30
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It is recommended at least 5 weeks after the initial repair when the residual inflammatory reaction in the area has subsided. The child can be placed on nasogastric (NG) feeds, and this also allows for respiratory symptoms to settle before surgery.4 The fistula should be cannulated (Fig. 1) at bronchoscopy to enable its identification during dissection.31–33 Depending on the level of the fistula an anterior neck crease or right thoracotomy is used to safely ligate the fistula, and suture the ends of the esophagus and trachea.4,34–37 Some have used a left extrapleural approach,38 a transcervical transtracheal approach,39 or via a median sternotomy.40 Many authors recommend placement of an interposition tissue between the sutured ends of the esophagus and trachea to prevent a second recurrence; these include mediastinal pleura,41–45 vascularized pedicle of pericardium,31,32,46 pedicled sternocleidomastoid flap,40 coastal cartilage graft,47 and omental flaps.48 The postoperative care is similar to after an ET.
Summary of Published Reports The outcomes after these two repairs are summarized in ►Tables 1–4. Overall, of the 57 patients managed using a variety of ETs there was an 84% (n ¼ 48) success rate compared with 93.5% (n ¼ 101) of 108 patients that had OS. Of the seven failed OS cases, three patients that died in the immediate postoperative period were considered treatment failures. One patient went on to have a cervical esophagostomy followed by gastric transposition,31 another two underwent an esophagectomy and gastric transposition each,32 and one
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366
3
1
Bhatnagar et al (1999)
Hoelzer et al (1999)c
European Journal of Pediatric Surgery
Khuranna et al (2004)
6
1
2
Wiseman (1995)
Lopes et al (2003)
1
Gutierrez et al (1994)
1
1
Vandenplas et al (1993)
McGahren et al (2001)
1
Al-Samarrai et al (1987)
1
2
Rangecroft et al (1984)
NG et al (1999)
n
References (23 articles)
13.6 (1–54)
11
3
–
72
2.6 (1–5)
25.5 (6–45)
0.5
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2.2 (1–3)
1
3
1
1
2.5 (1–4) (four attempts using laser)
2.5 (1–4)
1
4
1
5.5 (5–6)
Mean number of treatments required (range)a
1 (16%)
0
0
0
0
1 (33%) using electrocautery
Ongoing treatment in Px 1
0
0
0
0
Treatment failures converted to open surgery n (%)
53 (16 -96)
36
42
24
48
3
6 (Px 2 only)
3
12
24
12
Mean length of follow-up in mo (range)
Diathermy coagulation
n-BCA via bronchoscopy þ submucosal polidocanol via esophagoscopy.
De-epithelialization with cautery þ application of fibrin glue.
De-epithelialization by coagulation of fistula epithelium with holmium laser þ fibrin glue sealant.
Fibrin glue
(Continued)
Unspecified number of previous failed open repairs.
Three previous open repairs and one endoscopic gluing with fibrin failed.
Two previous failed endoscopic attempts with fibrin glue.
AL
Three AL
Electrocautery ¼ 2 Nd:YAG laser ¼ 1
AS; one previous failed open repair.
Two previous failed open repairs.
One previous failed open repair.
Notes
One previous failed endoscopic approach with electrocautery and n-BCA followed by failed thoracotomy with pleural flap. One AS. Respiratory distress in two using electrocautery
Postoperative complications
De-epithelialization with bronchial brush þ injection with fibrin glue.
Fibrin glue (Tisseel, Tissucol, Tisseal).
Fibrin glue; n-BCA; n-BCA þ submucosal 30% NaCl; n-BCA þ submucosal Polidocanol (Aethoxysklerol).
n-butyl cyanoacrylate (n-BCA, enbucrilate, Histoacryl) þ submucosal injection of 0.5% Polidocanol (Aethoxysklerol, Sclerovein) via esophagoscopy.
De-epithelialization (fulguration of fistula mucosa with diathermy).
Endoscopic technique (sealant þ/ de-epithelializationb)
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1 (16%)
1 (100%)
0
1 (100%)
1 (100%)
1 (33%) (using electrocautery)
1 (50%)
1 (100%)
0
1 (100%)
–
12
0
Successful single treatments n (%)
60 (1 Px)
Mean age at repair in mo (range)
Table 1 Summary of published endoscopic treatments of recurrent tracheoesophageal fistulas
Management of RTEF: A Systematic Review Aworanti, Awadalla
No. 5/2014
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European Journal of Pediatric Surgery
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3
1
4
3
1
1
1
Meier et al (2007)c
Keckler et al (2008)
Richter et al (2008) c
Sung et al (2008)
Yoon et al (2009)
Farra et al (2010)
Rakoczy et al (2010)
4
Linder (2006)
7
6
Tzifa et al (2005)
San Romàn et al (2006)
n
References (23 articles)
Table 1 (Continued)
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1 (100%)
– 1
1
1
3.3 (3–4)
1.2 (1–2)
3
2 (1–3)
1.5 (1–3)
4.3 (4–5)
1.6 (1–2)
Mean number of treatments required (range)a
0
0
0
0
0
0
1 (33%)
1 (14%)
1 (25%)
0
Treatment failures converted to open surgery n (%)
12
22
8
14 (9–22)
16
36
48 (?–60)d
89 (24–132)
45 (24–72)
66 (36–108)
Mean length of follow-up in mo (range)
Potassium titanyl phosphate laser
Submucosal injection of fibrin glue.
De-epithelialization with brush þ application of n-BCA þ Lipiodol glue via esophagus.
Chemocauterization using 50% trichloroacetic acid.
De-epithelialization with a Bugbee fulgurating diathermy electrode then sealed with fibrin glue þ added aprotinin.
Two previous failed endoscopic attempts with electrocautery þ fibrin glue.
AS
AS. Three previous failed open repairs.
One AS. Two previous failed open repairs.
One previous failed open repair with pleural interposition.
De-epithelialization þ plugging with a biosynthetic mesh (Surgisis) and fibrin glue at first attempt only
Notes
One previous failed open repair using a pleural interposition flap. Respiratory distress
One mortality case 3 days postoperatively. Cause bugbee electrocautery.
Postoperative complications
De-epithelialization with bronchial brush þ fibrin glue.
De-epithelialization by coagulation of fistula mucosa with diathermy in 4 Px þ fibrin glue sealant in all.
De-epithelialization with silver nitrate bead þ “ glue” and use of esophageal balloon to occlude airflow through fistula.
De-epithelialization by mechanical abrasion or bug by electrocautery followed by injection of n-BCA þ lipiodol into the fistula.
Endoscopic technique (sealant þ/ de-epithelialization b)
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1 (100%)
1 (100%)
0
3 (75%)
0
0.5
156
58 (3–156)
11.5 (3–20)
12
1 (33%)
3 (43%)
0
2 (33%)
Successful single treatments n (%)
Management of RTEF: A Systematic Review
39 (5–108)
0.55 (0.46–0.66)
18.9 (12–156)
16.5 (2–60)
Mean age at repair in mo (range)
368 Aworanti, Awadalla
5
1
57
Briganti et al (2011)
van Niekerk (2012)
Total/range
1 (100%)
21 (37%)
0.5–156e
0
Successful single treatments n (%)
6
7.8 (1–18)
Mean age at repair in mo (range)
2.2 (1–6) 106 procedures in 49 successful cases including 1 ongoing.
1
2
Mean number of treatments required (range)a
9 (16%)
0
3 (60%)
Treatment failures converted to open surgery n (%)
3–108e
De-epithelialization with endoscopic brush þ plugging with a porcine dermal biological mesh (Permacol) held in place by a synthetic absorbable sealant. Duraseal (polyethylene glycol þ trilysine amine)
De-epithelialization with biopsy forceps þ dextranomer/hyaluronic acid copolymer (Deflux) injected submucosaly.
–
24
Endoscopic technique (sealant þ/ de-epithelialization b)
Mean length of follow-up in mo (range)
Three (5%) respiratory distress; 1 (1.7%) mortality
Postoperative complications
Four (7%) previous leaks (AL); 10 (17%) previous strictures (AS); 16 previous failed open repairs; 6 previous failed alternative endoscopic Tx.
AS in all
Notes
European Journal of Pediatric Surgery
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Abbreviations: AL, anastomotic leaks before RTEF; AS, associated stricture before RTEF; mo, months; n, number; Px, patient; RTEF, recurrent tracheoesophageal fistula; Tx, treatment. Note: n-Butyl cyanoacrylate (n-BCA; Enbucrilate, Histoacryl); fibrin glue (Tisseel, Tissucol, Tisseal). a Mean number of treatments required for successfully treated cases. b De-epithelialization involves abrading, denuding, cauterizing, chemocauterizing, sclerosing, or destroying the epithelial lining of the fistula using bronchial brush, biopsy forceps, bead of silver nitrate, fulgurating diarthermy, electrocautery, potassium titanyl phosphate (KTP) laser, trichloroacetic acid (TCA), polidocanol (sclerosant), Nd:YAG laser, or holmium laser. c Literature review. d Median. e Reflects the range of available and compatible data only.
n
References (23 articles)
Table 1 (Continued)
Management of RTEF: A Systematic Review Aworanti, Awadalla
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369
European Journal of Pediatric Surgery
4
Girdany et al (1956)
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3 (100%) 3 (100%)
5.6 (2–11)
–
3
3
2
22
1
1
18
1
1
1
1
Kafrouni et al (1970) b
Stanford et al (1973)
Slim et al (1974)
Ein et al (1983)
Botham et al (1986)
Martin et al (1986)
Ghandour et al (1990)
Narasimhan et al (1990)
Vos et al (1996)
Holland et al (1998)
Delarue et al (2002)
0.5
11
90
0
2
1
1
1
1.2 (1–2)
1
1
1.2 (1–2)
1
0
0
0
0
1 (esophageal replacement)
0
0
0
0
0
0
1 (mortality)
1 (mortality)
0
1 (mortality)
0
Treatment failures converted to other procedure n (%)
27
4
Previous tracheal injury and AL. Subsequent planned esophageal reconstruction with colonic interposition.
Coastal cartilage graft þ fibrin sealant
AS
Nine AL
One previous failed open repair.
AL. One previous failed repair with pleural flap.
(9 of 23 Px) 39% AL; (10 of 23 Px) 43% AS.
Two AS and two AL. One Px also had stricture resection.
One AS
Three AS. One Px also had stricture resection.
Two AS
AS
Notes
Previous failed single open and multiple endoscopic attempts at repair.
Five leaks, 1 death; 10 mo postoperatively
Leak at the fistula site in four
One mortality
Esophageal leak þ esophagopleurocutaneous fistula. 1 mortality (patient died)
One mortality
Complications
Pedicled sternocleidomastoid flap via median sternotomy.
Pleural flap
Mediastinal pleura
– 144
Unspecified
Transcervical transtracheal repair
– –
Vascularized pedicle of pericardium
Simple division of fistula and suturing of ends
8
–
Left extra pleural approach
–
–
– Pleural flap
Closure with fine 6–0 monofilament stainless steel wire sutures.
–
–
“Tip of lung” interposed
0.5
Mediastinal pleura
Unspecified
– 12
Pleural flap
Surgical technique/ interposition tissue
5
Mean length of follow-up in mo (range)
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1 (100%)
1
1
14 (78%)
–
12
1
10
1
17 (77%)
–
5
2 (100%)
1
1
1
1
1
1.7 (1–2)
1
Mean number of treatments required (range)a
Management of RTEF: A Systematic Review
10.5 (5–16)
2 (66%)
5.3 (1–14)
3
Cowley (1967)
0
1 (100%)
1 (25%)
1 (100%)
Successful single treatments n (%)
1
3
23
5.75 (1–16)
24
Mean age at repair in mo (range)
Falletta (1964)
1
1
Hewlett (1955)
Moskovitz et al (1960)
n
References (21 articles)
Table 2 Summary of published open repairs of recurrent tracheoesophageal fistulas
370 Aworanti, Awadalla
1
2
4
26
11
108
Hotta et al (2002)
Bonnard et al (2007)
Briganti et al (2009)
Bruch et al (2010)
Coran (2013)
Total/range
4 (100%)
23 (88%)
6 (54%)
85 (79%)
–
–
0.5–90 d
2 (100%)
1
Successful single treatments n (%)
23.7 (12–30)
2.7 (1.7–3.6)
4
Mean age at repair in mo (range)
1.1 (1–2) 117 procedures in 101 successful cases
1.3 (1–2)
1.1 (1–2)
1
1
1
Mean number of treatments required (range) a
7 (6.5%; 3 early postoperative mortalities)
2 (both had esophagectomy and gastric transposition)
1 (cervical esophagostomy followed by gastric pull-up)
0
0
0
Treatment failures converted to other procedure n (%)
0.5–287d
–
–
17 (16%) leaks; 4 (3.7%) mortality
–
European Journal of Pediatric Surgery
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40 (37%) previous leaks (AL); 25 (23%) previous strictures (AS); 4 previous failed alternative open repair; 9 previous failed endoscopic Tx.
–
18 (68%) AS. Three had previous failed endoscopic Tx.
Seven leaks, four strictures.
84 (6–287)c Pleura n ¼ 11 Pericardium n ¼ 8 Muscle flap n ¼ 3 Lymph nodes n ¼ 2 No tissue n ¼ 1
–
–
One previous failed repair each. One endoscopic gluing and the other open using a pleural interposition flap. One AS.
Notes
Complications
Four AS. Three previous failed endoscopic Tx.
Omental flap interposition
Ligation and division of fistula
Surgical technique/ interposition tissue
(18–60)
20.5 (17– 24)
6
Mean length of follow-up in mo (range)
Abbreviations: AL, anastomotic leaks before RTEF; AS, associated stricture before RTEF; n, number; Px, patient; RTEF, recurrent tracheoesophageal fistula; Tx, treatment. Note: n-Butyl cyanoacrylate (n-BCA; Enbucrilate; Histoacryl); fibrin glue (Tisseel, Tissucol, Tisseal). a Mean number of treatments required for successfully treated cases. b Literature review. c Median. d Reflects the range of available and compatible data only.
n
References (21 articles)
Table 2 (Continued)
Management of RTEF: A Systematic Review Aworanti, Awadalla
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371
Management of RTEF: A Systematic Review
Aworanti, Awadalla
Table 3 Summary of published outcomes of endoscopic treatment versus open surgery for recurrent tracheoesophageal fistulas Descriptive data
Endoscopic treatment
Open surgery
Number of patients
57
108
Age range at treatment onset in moa
0.5–156
0.5–90
Successful single treatments n (%)
21 (37%)
85 (79%)
Mean number of treatments required (range)
2.1 (1–6)
1.1 (1–2)
Treatment failures converted to other procedure n (%)
9 (16%) (84% overall success)
7 (6.5%) (93.5% overall success including three perioperative mortalities)
Mean length of follow-up in mo (range)a
3–108
0.5–287
Abbreviation: mo, months. a Range for available data only.
Table 4 Summary of results comparing de-epithelialization only versus sealant only versus de-epithelialization þ sealant Descriptive data
De-epithelialization only (5 articles)
Sealant only (4 articles)
De-epithelialization þ sealant (15 articles)
Number of patients
15
6
36
Successful single treatments n (%)
3 (20%)
4 (67%)
14 (39%)
Mean number of treatments performed (range)
2.7 (1–6)
1.5 (1–3)
2 (1–5)
Treatment failures converted to other procedure n (%)
2 (13%)
1 (16.6%)
6 (16.6%)
Note: When multiple techniques were used on one patient the last technique described was used to deduce the final outcome.
had an unspecified esophageal replacement.37 The refistulation rate after OS was 21% and an average of 1.1 (range 1–2) procedures were required to achieve treatment success. However after ET, treatment was either incomplete or resulted in refistulation in 63%, and though only an average of 2.1 treatments were required to achieve successful closure, occasionally up to 6 treatments were necessary.15 Both averages excluded several treatment attempts in unsuccessful cases that were subsequently converted to an alternative treatment modality; the failed ET cases were successfully treated by OS. These results were reported after a maximum follow-up of 9 years and 23 years for ET and OS, respectively. The major complications after OS were 17 (16%) leaks and 4 (3.7%) deaths; 1 of these deaths occurred 10 months after surgery and a second RTEF was found at autopsy.37 These mortalities after OS were reported in 1956, 1964, 1967, and 1990. After ET, three suffered significant respiratory distress postoperatively and there was 1 (1.7%) death reported in 2005; this occurred 3 days postprocedure and the cause was unspecified.24 The results when ET were grouped into “de-epithelialization only,” “sealant only,” and “de-epithelialization combined with sealants” (as suggested by Richter et al28) is summarized in ►Table 4. There were no significant observations except that contrary to Richter’s findings, the use of “sealants only” resulted in the most successful single treatments reported (67%) and the least number of treatments required (mean 1.5; range 1–3). This finding should however be interpreted with caution due to the small numbers, particularly in the “sealant only” group (n ¼ 6). European Journal of Pediatric Surgery
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Discussion Apart from inadequate ligation in the first instance, these fistulas probably recur due to an anastomotic leak that forms a localized abscess which bores into the trachea at the point of least resistance: the suture lines. Strictures that require forceful dilatations can lead to the same phenomenon.26,32,34,36,46 This review suggest that one in four and one in five cases of RTEF reported in the literature document an association with leaks or strictures, respectively. The mean time to recurrence is 2 months,4,31 but have been reported up to 32 years after the initial repair.49 The usual clinical presentation includes persistent coughing due to saliva aspiration, choking, and apnea during feeds, recurrent aspiration
Fig. 1 An operative view via an anterior neck dissection showing easy access to the trachea (a), the cannulated fistula (arrow), and the retracted esophagus (b).
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372
pneumonia, and eventually chronic lung disease.4,24,28,37 A prone esophagram performed by injecting the contrast under pressure via an NG tube while withdrawing the tube is the most sensitive investigation with the least false negatives.4,35,50 The diagnosis is confirmed at bronchoscopy or esophagoscopy occasionally requiring the aid of methylene blue.7,26,31,32 Important differentials are tracheal diverticula51 or a missed second fistula.52,53 When a RTEF has being diagnosed, OS was the only management option available until 1974 when ET was first described by Gdanietz et al.54 Since then there has been a variety of endoscopic techniques with varying results published. The drive to develop and improve these new techniques stem from the belief that open redo surgery is technically challenging and plagued with a high morbidity, mortality, and further recurrences. Most authors that quote these poor expectations directly or indirectly reference Kafrouni et al review from19707 where they reported a 59% mortality after RTEF.10–14,20,24,26,28,55 However, on further analysis of their review, the actual mortality after redo surgery for RTEF was 32% (9 of 28 reoperated cases). Manning et al as early as 1986 has reported a fall in operative mortality rates after primary TEF repair from 56 to 6.9%, since Haight’s pioneering repair performed at their center5; one would then expect that the mortality after surgery for RTEF should show a similar improvement. Our review of the published reports of OS for RTEF from 1955 to 2013 revealed only three perioperative deaths that all occurred in the 60s. The major complications after OS were leaks and second recurrences; another redo surgery for a second recurrence will be challenging for obvious reasons. In this review, a second recurrence (after OS) was reported in 21% of the reported RTEF cases, comparable to 22, 22, and 11.5% reported in the OS patient series by Ein et al (1983),4 Ghandour et al (1990),37 and Bruch et al (2010),31 respectively. The use of a vascularized interposition tissue is recommended to prevent a second RTEF. No more than two OS treatments are usually required and treatment failures resulted in some form of esophageal replacement reported in only four patients. Hence, this updated review shows that OS can be safely performed without a high morbidity and mortality. Minimally invasive techniques are widely accepted to be better tolerated by patients and whenever possible such options should be explored, thus supporting the case for ET; however, the outcomes should be comparable. Several authors have used a variety of techniques for ET, summarized above, all hoping to achieve better outcomes. There have been three literature reviews on ET. In Hoelzer 1999 review,12 15 cases were reported, having been treated with diarthermy, laser, n-BCA, and fibrin glue, and 1 to 5 treatments were necessary with complete closure in only 10 patients (67%). Meier et al in 200726 reported an overall success rate of 60% of 62 patients culled from the literature (fibrin glue 55% [n ¼ 22], n-BCA 48% [n ¼ 29], laser 80% [n ¼ 5], diathermy 50% [n ¼ 10], n-BCA þ sclerosant100% [n ¼ 5]); the mean number of applications ranged from 1 to 5.5. They concluded that ET is safe, effective, has less morbidity than OS and recommended fibrin glue but advised close follow-up. Richter et al28 in their 2008 review, culled 37 patients from the literature and categorized ET into: application of sealant only
Aworanti, Awadalla
(n ¼ 14; 78.6% success), de-epitheliazation of the fistula tract only (n ¼ 8; 62.5% success), de-epitheliazation combined with sealant (n ¼ 15; 93.3%). Overall there was an 81.1% success rate (48.6% after a single attempt) and they concluded that deepitheliazation with diathermy combined with fibrin glue sealant will yield the best results; fibrin glue by nature would work better if the opposing surfaces are closely apposed and not epithelialized.21 The failures seen with ET may be partly because the sealants fibrin glue and n-BCA are biodegradable. Aprotinin, an antifibrinolytic, prolongs the stability of fibrin glue in vivo for up to 10 days only56; however, the fibrin clot formed has a loose scaffold that encourages in-growth of fibroblasts. n-BCA, commonly used for skin closure, is not recommended for mucosal application by most manufacturers, as its stability and possible absorption in such environments is not yet fully determined. Willets et al (1998)55 also appreciating the possibility of late refistulation after ET, and the limited follow-up period of the reported cases managed endoscopically, decided to evaluate the long-term outcome of these patients by contacting the respective authors. They concluded that of 22 patients initially reported to have had successful ET, only 12 (55%) remained closed after a median follow-up of 107 months; there was however no morbidity or mortality directly related to these procedures. These findings somewhat mirror ours (84% overall success after a range of 1–6 ET). However, there were three reports of significant respiratory distress following ET; aspiration of the sealant or perforation of the tracheal wall when using diathermy or laser for de-epitheliazation are serious potential hazards.8,23,29 Furthermore, these patients that require up to six ETs suggest that they are representing with the typical symptoms of saliva and feed aspiration that, aside from the burden to the family of frequent hospitalization, can eventually lead to chronic lung disease. Unless there is a systematic way of evaluating for refistulation after ET, lung morbidity may be unavoidable. On the other hand, no child required more than two open operations for RTEF, and anastomotic leaks, usually managed conservatively, were the major complications to contend with. The main limitations of this review include the heterogeneity of the included articles, particularly as case reports would predominantly report positive outcomes. Furthermore, some authors occasionally mentioned a variety of previous failed OS and/or ET attempts in the patients whom they formally published their definitive repair on; these previous repairs (due to insufficient data on them) could not be considered when analyzing our results. However, they are mentioned in ►Tables 1 and 2.
Conclusion In conclusion, based on our findings on 165 published cases, OS for the repair of recurrent TEF has a low morbidity, mortality, and re-fistulation rate particularly when an interposition tissue is placed between the trachea and esophagus. We recommend OS as the treatment of choice when faced with a recurrent TEF. Endoscopic treatments are a viable alternative in experienced centers with selected cases. Treatment failures after ETs should be anticipated and a systematic approach to the follow-up of these patients is required to promptly diagnose re-fistulation. European Journal of Pediatric Surgery
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Management of RTEF: A Systematic Review
Management of RTEF: A Systematic Review
Aworanti, Awadalla
Further clinical trials and controlled experiments are necessary to devise the technique and ideal, preferably nonbiodegradable, sealant that guarantees a definitive endoscopic closure of a recurrent TEF after one attempt.
19
20 21
Conflict of Interest None.
22
References
23
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3 4
5
6
7 8 9
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12
13
14
15
16
17
18
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50 Stringer DA, Ein SH. Recurrent tracheo-esophageal fistula: a
protocol for investigation. Radiology 1984;151(3):637–641 51 Shah AR, Lazar EL, Atlas AB. Tracheal diverticula after tracheoe-
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European Journal of Pediatric Surgery
Vol. 24
No. 5/2014
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41 Hewlett TH. Recurrent tracheo-esophageal fistula; case report.
Aworanti, Awadalla
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