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Author's personal copy International Journal of Pediatric Otorhinolaryngology 78 (2014) 985–989
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International Journal of Pediatric Otorhinolaryngology journal homepage: www.elsevier.com/locate/ijporl
Review Article
Cervical/thoracotomic/thoracoscopic approaches for H-type congenital tracheo-esophageal fistula: A systematic review Filippo Parolini a,b,*, Anna Morandi a, Francesco Macchini a, Valerio Gentilino a, Andrea Zanini a, Ernesto Leva a a b
Department of Paediatric Surgery, Fondazione IRCCS Ca’ Granda, Ospedale Maggiore Policlinico, Milano, Italy Department of Paediatric Surgery, Azienda Ospedaliera Spedali Civili Brescia, Italy
A R T I C L E I N F O
A B S T R A C T
Article history: Received 2 March 2014 Received in revised form 2 April 2014 Accepted 4 April 2014 Available online 5 May 2014
Purpose: Aim of this systematic review is to investigate the thoracic and cervical surgical approaches of H-type tracheo-esophageal fistula (TEF) according to the position of the fistula. Methods: The PubMed database was searched for original studies on H-type TEF treatment published between 1977 and 2012. Manuscripts finally included were divided into open and thoracoscopic surgery groups. Results: Seventeen studies were selected for open surgery group, and most of them agree on the importance of pre-operative diagnosis of the fistula by preliminary tracheoscopy. Right cervicotomy was used in 70 cases (76.9%), left cervicotomy in 12 (13.2%), and thoracotomy only in 9 (9.9%). Five studies were included in thoracoscopic group (6 patients). Indications for the surgical approach (cervical vs thoracic) according to the position of the TEF were clearly described in 10 manuscripts, and all stated differences in surgical technique details. Complications and mortality rates were not statistically correlated to the different surgical approaches. Conclusions: The evidence base in regard to the treatment of H-type fistula in children is poor and the skills and preferences of the surgeons guide the choice of the procedure. Surgical division of the fistula is curative, and the key to a successful repair is the pre-operatively identification of the level of the fistula with tracheoscopy. Right cervicotomy seems to be the approach of choice in the majority of case, with the thoracic approach appropriate only for fistulae opening below T2. Further well-designed prospective studies which take into account of selection and performance bias are strongly required. ß 2014 Elsevier Ireland Ltd. All rights reserved.
Keywords: Tracheo-esophageal fistula Cervicotomy Thoracotomy Thoracoscopy
Contents 1. 2.
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Introduction . . . . . . . . . . . . . . . . . . . . . . . . . . . . Methods . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . 2.1. Data sources . . . . . . . . . . . . . . . . . . . . . . 2.2. Study selection . . . . . . . . . . . . . . . . . . . . 2.3. Data extraction and quality assessment Results . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . 3.1. Description of studies . . . . . . . . . . . . . . . 3.2. Open surgery group . . . . . . . . . . . . . . . . 3.3. Thoracoscopic surgery group . . . . . . . . . Discussion . . . . . . . . . . . . . . . . . . . . . . . . . . . . . References . . . . . . . . . . . . . . . . . . . . . . . . . . . . .
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Abbreviation: TEF, tracheo-esophageal fistula. * Corresponding author at: Department of Paediatric Surgery, Fondazione IRCCS Ca’ Granda, Ospedale Maggiore Policlinico, Padiglione Alfieri (Chirurgia Pediatrica), Via Commenda 10, 20122 Milano, Italy. Tel.: +39 02 55032545; fax: +39 02 55036570. E-mail address: parfi
[email protected] (F. Parolini). http://dx.doi.org/10.1016/j.ijporl.2014.04.011 0165-5876/ß 2014 Elsevier Ireland Ltd. All rights reserved.
Author's personal copy F. Parolini et al. / International Journal of Pediatric Otorhinolaryngology 78 (2014) 985–989
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1. Introduction Tracheo-esophageal fistula (TEF) without esophageal atresia (H-type TEF) is a rare congenital malformation that accounts for about 4% of tracheo-esophageal malformations and has an incidence of around 1:50,000–80,000 births [1]. Surgical division of the fistula is curative, and the key to a successful repair is the pre-operatively identification of the level of the fistula with tracheoscopy, usually performed in collaboration with pediatric otorhinolaryngologists [2]. Although there is a general agreement that in the majority of H-type TEF cervicotomy is the approach of choice [3], when the fistula is located near the border between the neck and the thorax choosing the appropriate approach, cervical or thoracic is difficult. We therefore undertook a systematic review of the available case reports, case series and reviews on H-type TEF treatment, in order to evaluate the effectiveness of the different surgical approaches according to the position of the fistula. 2. Methods 2.1. Data sources For this systematic review we adhered to PRISMA guidelines [4,5].
[(Fig._1)TD$IG]The PubMed database (http://www.ncbi.nlm.nih.gov/pubmed/) was
searched for studies on treatment of H-type TEF that were published since January 1977. The following keywords were searched individually or in association: isolated trachea-esophageal fistula, H-type tracheo-esophageal fistula, N-type tracheo-esophageal fistula, cervicotomy, thoracoscopy and thoracotomy. The date of the last search was December 2012. 2.2. Study selection Inclusion criteria were English and French articles that reported original data on treatment of newborn affected by tracheaesophageal fistula without esophageal atresia. Eligible study designs were cohort, case report, case series and review. The study selection process is presented in Fig. 1. We omitted reports in which titles or abstracts indicated that they were not human studies, they encompassed TEF associated to esophageal atresia, recurrent TEF and acquired TEF, they reported endoscopic treatment of TEF and they not clearly reported the method of treatment. Duplicated studies were also removed. We then evaluated the full text of the passed articles. Titles and abstracts of identified publications were checked and reviewed against the predefined inclusion criteria, and afterward, the full text the articles were similarly assessed for eligibility. Manuscripts finally included in the study were divided into two categories: open and thoracoscopic surgery.
Fig. 1. Identification of studies from PubMed databases and inclusion in the systematic review.
Author's personal copy F. Parolini et al. / International Journal of Pediatric Otorhinolaryngology 78 (2014) 985–989
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Table 1 Open surgery group systematic review. Primary outcomes
Secondary outcomes
Period
n
Right cervicotomy
Left cervicotomy
Thoracotomy
Endoscopic study
Cannulation of the fistula
Level of the fistula
Complications and mortality
Atzori [8] Babu [9] Bakhos [10] Biechlin [11] Brookes [12] Crabbe [13]
97-03 2005 97-02 98-06 85-05 85-95
2 1 3 8 7 16
2 1 3 8 7 16
0 0 0 0 0 0
0 0 0 0 0 0
Yes Yes Yes Yes Yes Yes
Yes No No No Yes Yes
3.5F Fog – – – 3–4F Fog NA
NA T2–T3 NA NA C5–T3 NA
Garcia [2] Genty [14] a
91-97 89-97
4 8a
4 2a
0 6
0 0
Yes Yes
Yes Yes
Guide wire Ureteral stent
NA 1.5–4 cm above carina
Goyal [15] Kane [16] Karnak [17] Ko [18] LaSalle [19] Mattei [20] b Riazulhaq [21] Sahnoun [22] Yazbeck [23]
02-04 2007 71-96 2000 74-77 2012 2011 01-10 61-81
3 1 10 1 4 1 1 4 16
3 1 4 0 4 1 1 1 12
0 0 5 1 0 0 0 0 0
0 0 1 0 0 1 0 3 4
Yes Yes Yes Yes Yes Yes No Yes No
Yes Yes Yes Yes NA Yes No NA Yes
3F ureteric cath. 3F ureteric cath. Ureteral cath. Guidewire – Fog – – Fog
NA Thoracic inlet NA T1–T2 NA Above carina T1 T2–T4 C7–T4
None None None Tracheomalacia (1) None Recurrent fistula (2), stricture (3) None Stricture (1), left recurrent laryngeal paralysis (1) None None None None None None None Death (2) Recurrent fistula (1), death (1)
Total n (%). Right cervicotomy: 69 (76.7). Left cervicotomy: 12 (13.3). Anterior cervicotomy: 1 (1.0). Thoracotomy: 9 (10). Legends: cath. = catheter, Fog = fogarty, and NA = not available data. a The first patients of this series underwent an anterior cervicotomy. b Two H-type fistulas were present in one child.
2.3. Data extraction and quality assessment Two independent authors extracted information related to the study. Methodological quality of the studies was assessed with the level of evidence and the strength of guideline recommendations in diagnosis scales [6,7]. For each study, data were extracted for three primary outcomes and many secondary outcomes. The primary outcomes were the number of treated patients, the study duration and the surgical approach. For open surgery group, the secondary outcomes were the level of the fistula, the use of contrast studies, the use of endoscopy, the cannulation of the fistula, the complications and the mortality. For thoracoscopic series, secondary outcome were the level of the fistula, the complications and the mortality, and many surgical details. Categorical variable frequencies where by using Pearson’s chisquare test (x2) or two-tailed Fisher exact probability test. A Pvalue
