Journal of Pediatric Surgery xxx (2014) xxx–xxx

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Thoracoscopic repair of esophageal atresia with tracheoesophageal fistula: Overcoming the learning curve Sanghoon Lee, Suk-Koo Lee, Jeong-Meen Seo ⁎ Department of Surgery, Samsung Medical Center, Sungkyunkwan University School of Medicine, Seoul, Korea

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Article history: Received 27 January 2014 Received in revised form 11 March 2014 Accepted 27 April 2014 Available online xxxx Key words: Esophageal atresia Tracheoesophageal fistula Thoracoscopy Minimally invasive

a b s t r a c t Background/Purpose: We describe our initial experience of thoracoscopic esophageal atresia with distal tracheoesophageal fistula (EA/TEF) repair. Methods: Twenty-three consecutive cases of thoracoscopic repair of EA/TEF were performed between October 2008 and March 2013. The medical records of 22 patients were retrospectively reviewed, excluding one case of early postoperative mortality. Results: The study period was divided into two sections: from 2008 to 2011 (13 cases) and from 2012 to 2013 (9 cases). Patient characteristics did not differ between the two study periods. Mean operation time was significantly shorter in period 2 compared to period 1 (p b 0.01). There was one case that was converted to open repair via thoracotomy during period 1. Anastomosis leakage was seen in two cases during period 1, while no cases of leakage were encountered in period 2. Ten of 13 cases (76.9 %) in period 1 had clinically significant esophageal strictures requiring one or more sessions of balloon dilatations. No cases of esophageal strictures were seen in period 2 (p b 0.01). Conclusions: When the initial learning curve was endured, surgical outcomes of thoracoscopic repair of EA/TEF were superior to the outcomes of earlier cases, and comparable to past experiences with open thoracotomy. © 2014 Elsevier Inc. All rights reserved.

Minimally invasive approach is becoming more widely applied to areas of pediatric surgery, including diseases previously considered difficult even with conventional methods, such as neonatal surgery. The minimally invasive technique for surgical correction of esophageal atresia (EA) was first described by Lobe et al., and has since been gaining acceptance among many pediatric endoscopic surgeons [1–3]. The thoracoscopic repair of EA remains a considerable challenge, even for the most experienced pediatric surgeon. Nonetheless, a survey of pediatric surgeons in the United Kingdom showed that 46% of those surveyed hoped to implement this procedure in their practice [4]. An endoscopic procedure of such complexity requires much trialand-error before it is adequately set up, thus reports on the experience of early trials from centers that have now gained expertise would provide crucial knowledge to these surgeons seeking to start up their own program of thoracoscopic EA repair. Here we describe our initial experience of thoracoscopic esophageal atresia with distal tracheoesophageal fistula (EA/TEF) repair, focusing on the changes in perioperative results and short-term outcome.

⁎ Corresponding author at: Department of Surgery, Samsung Medical Center, Sungkyunkwan University School of Medicine, 50 Irwon-dong, Gangnam-gu, Seoul 135–710, Korea. Tel.: +82 2 3410 0282; fax: +82 2 3410 0040. E-mail address: [email protected] (J.-M. Seo).

1. Patients and methods The first case of thoracoscopic repair of EA/TEF was performed in October 2008 at our center (Samsung Medical Center, Seoul, Korea). Since then, thoracoscopic repair was the surgical method of choice for all infants with EA/TEF. Twenty-three consecutive cases of thoracoscopic repair of EA/TEF were performed between October 2008 and March 2013. During this period, one case of pure esophageal atresia and one case of tracheoesophageal fistula without esophageal atresia were also surgically corrected by thoracoscopic approach. All cases of thoracoscopic EA/TEF repair were performed by a single surgeon (J.M.S.) experienced in neonatal laparoscopic and thoracopic surgery. The medical records of these 23 patients were retrospectively reviewed. After general anesthesia, the infant is placed in a semi-lateral position facing left and 45° downwards, with its right arm raised up to the head to expose the right axillary region. A 5 mm trocar is inserted just below the right scapula tip and a 5 mm camera is inserted. A 3 mm trocar is inserted at the right mid-axillary line along the scapula margin and another 3 mm trocar is inserted opposite the 5 mm trocar. The conformation of the three trocars is placed so as to form a straight line, with the two 3 mm trocars (working ports) at same distances from the 5 mm trocar. Carbon dioxide is instilled at flow rate of 3 mL/min and up to a maximum pressure of 5 mmHg to collapse the lung and allow good visualization of the esophagus and trachea. When the infant shows stable vital signs and oxygen saturation, the procedure is begun.

http://dx.doi.org/10.1016/j.jpedsurg.2014.04.016 0022-3468/© 2014 Elsevier Inc. All rights reserved.

Please cite this article as: Lee S, et al, Thoracoscopic repair of esophageal atresia with tracheoesophageal fistula: Overcoming the learning curve, J Pediatr Surg (2014), http://dx.doi.org/10.1016/j.jpedsurg.2014.04.016

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S. Lee et al. / Journal of Pediatric Surgery xxx (2014) xxx–xxx

The azygos vein is divided with electrocautery. The distal end of the esophagus is identified and dissected in a proximal direction to the trachea. The tracheoesophageal fistula is ligated by suture ligation with 3-0 braided polyglycolic acid sutures (Safil®, B. Braun, Melsungen, Germany). Anastomosis is done by interrupted sutures using 5-0 glyconate monofilament sutures (Monosyn, B. Braun, Melsungen, Germany). A 10 Fr chest tube is routinely inserted with the tip placed near the esophageal anastomosis. Postoperatively, the infant is initially kept on mechanical ventilation in the intensive care unit. However, ventilator weaning is attempted as soon as possible. An 8 Fr feeding tube is kept in place and oral intake is restricted for 7 days. On the seventh postoperative day, contrast esophagography is performed and when there is no sign of leakage from the anastomosis site, oral intake is initiated and the chest tube is removed the following day. Mann–Whitney tests were used to compare continuous variables and chi-square tests or the Fisher’s exact test, when necessary, were used to compare categorical variables. All continuous variables were of non-normal distributions from the Shapiro–Wilk test. p-Values of b 0.05 were considered to be significant.

2. Results From October 2008 to March 2013, 23 infants were diagnosed with EA/TEF at Samsung Medical Center. Twenty-three consecutive cases of thoracoscopic EA/TEF repair were done in these infants during this period. One infant died at postoperative 22 days because of complications related to its cardiac anomaly, and was excluded from the analysis. The study period was divided into two sections: from 2008 to 2011 (13 cases) and from 2012 to 2013 (9 cases). Patient demographics are shown in Table 1. Patient characteristics did not differ between the two study periods, in terms of gestational age, birth weight, age at operation, body weight at operation and rate of associated anomalies. Median follow-up was 26 months (range 3-56 months). Table 2 outlines the surgical outcome between the two study periods. Mean operation time was significantly shorter in period 2 compared to period 1 (p b 0.01). There was one case that was converted to open repair via thoracotomy during period 1. Postoperative use of mechanical ventilation was significantly shorter in period 2 and length of postoperative hospital stay was significantly shorter in period 2 (p b 0.05). Days required to reach full oral feeding was shorter in period 2, although not statistically significant. Radiologic evidence of anastomosis leakage was seen in two cases during period 1, while no cases of leakage were encountered in period 2. Ten of 13 cases (76.9 %) in period 1 had clinically significant esophageal strictures requiring one or more sessions of balloon dilatations. Median number of balloon dilatations performed in these 10 patients was 5 (range 1-7). No cases of esophageal strictures were seen in period 2 (p b 0.001). Similar number of gastroesophageal (GE)

Operation time (minutes, mean ± SD) Conversion to open surgery (%) Mechanical ventilation (postoperative days, mean ± SD) Full oral feeding (days, mean ± SD) Hospital stay (postoperative days, mean ± SD) Anastomosis leakage (%) Strictures requiring balloon dilatation (%) Balloon dilatations (median) Gastroesophageal reflux (%) Fundoplication (%)

Overall

Period 1 (2008-2011, n = 13)

Period 2 (2012-2013, n = 9)

p-Value

179 ± 81

215.6 ± 83.5

118.3 ± 30.2

0.001

1 (0.5)

1 (8)

0



3.8 ± 3.7

5.0 ± 4.4

2.1 ± 1.1

0.039

15.0 ± 6.5

17.1 ± 7.0

12.0 ± 4.6

0.054

26.3 ± 19.9

33.7 ± 22.7

15.6 ± 6.5

0.016

2 (0.9) 10 (45.4)

2 (15.4) 10 (76.9)

0 0

0.494 b0.001

5 (range 1-7) 5 (range 1-7) – 8 (36.3) 5 (38.5) 3 (33.3) 5 (22.7) 5 (38.5) 0

– 0.827 0.054

reflux was diagnosed in each period, and all five cases in period 1 were surgically managed with fundoplication. The characteristics and treatment outcome of the 10 patients with clinically significant esophageal strictures are shown in Table 3. Five out of 10 patients had radiologic evidence of anastomosis stricture on esophagography done in the immediate postoperative period. Symptoms of esophageal stricture became evident early in the majority of patients with eight cases requiring their first balloon dilatation before 4 months. Outcome of treatment for esophageal stricture was good in eight patients (8/10, 80%), including one patient who had symptom dissolution after esophagoesophagostomy revision owing to an esophageal diverticulum. 3. Discussion For all types of surgery, a learning curve exists before the surgeon is able to acquire the adequate skills and experience required to tactfully carry out the surgical procedure. This learning curve becomes more evident for endoscopic procedures; especially in case of a highly advanced endoscopic procedure such as thoracoscopic EA/TEF repair, a learning curve has to be taken into account, even for the most experienced endoscopic surgeon [5,6]. In our center, we performed the first case of thoracoscopic EA/TEF repair in 2008. Since then, we have used the thoracoscopic approach in all of our EA/TEF patients and have accumulated 23 consecutive cases. Upon retrospective review of our experience, we were able to observe a steady decline in operative time during the entire period spanning over 4 years. Also, during the latter part of our experience, we have seen a significant decline in postoperative complications, possibly reflecting the learning curve for this surgical procedure. Table 3 Outcome of postoperative esophageal anastomosis stricture.

Table 1 Patient demographics. Overall

Male/Female Gestational age (weeks, mean) Birth weight (g, mean ± SD) Age at operation (days, median) Weight at operation (g, mean ± SD) Associated anomaly (%)

Table 2 Study outcome.

Period 1 (2008-2011, n = 13)

Period 2 (2012-2013, n = 9)

p-Value

13:9 37+2

5: 8 36+4

8: 1 38+1

0.031 0.183

2627 ± 509

2609 ± 572

2653 ± 433

0.845

2 (range 1-28)

6.4

2.6

0.116

2630 ± 510

2657 ± 441

2593 ± 386

0.699

16 (72.7)

9 (69.2)

7 (77.8)

0.869

Case no. Period Age at first BD Number of BDs GER Fundoplication Outcome 1 2 3 4 5 6 7 8 9 10

1 1 1 1 1 1 1 1 1 1

3 months 1 months 15 months 13 months 2 months 2 months 2 months 2 months 3 months 4 months

6 7 1 2 1 5 5 2 6 5

Yes Yes No No No Yes No Yes No Yes

Yes Yes No No No Yes No Yes No Yes

Improved Improved Improved Improved Improved Improved Persistent Improved Persistent Improveda

BD, balloon dilatation; GERD, gastroesophageal reflux. a After esophagoesophagostomy revision.

Please cite this article as: Lee S, et al, Thoracoscopic repair of esophageal atresia with tracheoesophageal fistula: Overcoming the learning curve, J Pediatr Surg (2014), http://dx.doi.org/10.1016/j.jpedsurg.2014.04.016

S. Lee et al. / Journal of Pediatric Surgery xxx (2014) xxx–xxx

Our analysis was done by comparing the results of thoracoscopic EA/TEF repair between period 1 (2008-2011) and period 2 (2012-2013). As shown in Table 1, there were no differences in patient characteristics between the two periods (albeit a higher proportion of female patients in period 2). However, mean operation time was significantly shorter in period 2 compared to period 1. The longer operation time during period 1 may be attributable to the earlier problems encountered because of our lack of experience in the optimal placement of trocars, the surgeon’s lack of familiarity with the procedure, and difficulties in anesthetic management. Van der Zee et al. reported the results of thoracoscopic EA repair, comparing their earlier 41 cases with their later 31 cases [5]. The mean operation time remained unchanged, and they claimed that this was because of cases in the later part of their series being performed by junior staff members less experienced in the procedure. All cases in our study were performed by a single surgeon, and the mean operation time in period 2 was significantly shorter compared to period 1. Overall, there was only one case that required conversion to open thoracotomy, which was our third case performed. However, the choice for open conversion in this case was not surgery-related. The infant was not able to sustain adequate oxygen saturation after CO2 insufflation and subsequent collapse of the right lung. The overall anastomosis stricture rate in our series is 45.4 %, which is comparable to previous reports [3,4,7]. However, we were able to observe a sharp decline in anastomosis stricture, as well as leakage in period 2 compared to period 1. We have not made major modifications to our anastomosis technique during the study period and there was a similar rate of GE reflux between the two periods. Thus the decline in strictures is likely because of the gain in experience and expertise in the thoracoscopic procedure by the operating surgeon. Minor changes in the operative technique were, minimal handling and dissection of the distal esophagus during preparation for the esophageal anastomosis and using the 3.5 mm Hook Scissors (Aesculap, Tuttlingen, Germany) for more precision during opening of the proximal esophageal stump. Esophageal stricture was managed with intermittent esophageal balloon dilatations, based on symptoms and radiologic evidence. Two patients have persistent strictures after five and six sessions of balloon dilatation, respectively, without evidence of GE reflux. Surgical management of GE reflux by fundoplication was performed in all

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five cases of GE reflux diagnosed in period 1, while all cases of GE reflux in period 2 were not surgically treated. This was because of our policy of surgically treating GE reflux only when there is clinical evidence of anastomotic stricture, in which case the reflux may hinder the management of the anastomotic stricture [8,9]. We did not experience any recurrence of tracheoesophageal fistula among our 22 cases. Previous studies have reported 1.9 to 4% fistula recurrence rates [3,5,7]. These reports describe their method of fistula closure as either suture ligation or application of a surgical clip. We have uniformly used suture ligation with 3-0 braided polyglycolic acid sutures for fistula ligation in all of our cases. The absence of recurrent tracheoesophageal fistula in our series may be attributed to the suture ligation technique. In conclusion, the initial period of learning curve was evident in our earlier 13 cases of thoracoscopic EA/TEF repair. However, once this period of learning curve was endured, surgical outcomes were superior to the outcomes of earlier cases, and comparable to past experiences with open thoracotomy. References [1] Lobe TE, Rothenberg S, Waldschmidt J, et al. Thoracoscopic repair of esophageal atresia in an infant: a surgical first. Pediatr Endosurg Innov Tech 1999;3:141–8. [2] Al Tokhais T, Zamakhshary M, Aldekhayel S, et al. Thoracoscopic repair of tracheoesophageal fistulas: a case-control matched study. J Pediatr Surg 2008;43:805–9. [3] Holcomb III GW, Rothenberg SS, Bax KM, et al. Thoracoscopic repair of esophageal atresia and tracheoesophageal fistula: a multi-institutional analysis. Ann Surg 2005;242:422–8 [discussion 428–30]. [4] Ron O, De Coppi P, Pierro A. The surgical approach to esophageal atresia repair and the management of long-gap atresia: results of a survey. Semin Pediatr Surg 2009;18:44–9. [5] van der Zee DC, Tytgat SH, Zwaveling S, et al. Learning curve of thoracoscopic repair of esophageal atresia. World J Surg 2012;36:2093–7. [6] Szavay PO, Zundel S, Blumenstock G, et al. Perioperative outcome of patients with esophageal atresia and tracheo-esophageal fistula undergoing open versus thoracoscopic surgery. J Laparoendosc Adv Surg Tech A 2011;21:439–43. [7] Lugo B, Malhotra A, Guner Y, et al. Thoracoscopic versus open repair of tracheoesophageal fistula and esophageal atresia. J Laparoendosc Adv Surg Tech A 2008;18:753–6. [8] van der Zee DC, Bax KN. Thoracoscopic treatment of esophageal atresia with distal fistula and of tracheomalacia. Semin Pediatr Surg 2007;16:224–30. [9] Koivusalo A, Pakarinen MP, Rintala RJ. The cumulative incidence of significant gastrooesophageal reflux in patients with oesophageal atresia with a distal fistula—a systematic clinical, pH-metric, and endoscopic follow-up study. J Pediatr Surg 2007;42:370–4.

Please cite this article as: Lee S, et al, Thoracoscopic repair of esophageal atresia with tracheoesophageal fistula: Overcoming the learning curve, J Pediatr Surg (2014), http://dx.doi.org/10.1016/j.jpedsurg.2014.04.016

Thoracoscopic repair of esophageal atresia with tracheoesophageal fistula: overcoming the learning curve.

We describe our initial experience of thoracoscopic esophageal atresia with distal tracheoesophageal fistula (EA/TEF) repair...
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