Pediatr Surg Int (2014) 30:883–887 DOI 10.1007/s00383-014-3554-2
ORIGINAL ARTICLE
Thoracoscopic versus open repair of esophageal atresia with tracheoesophageal fistula at a single institution Masaya Yamoto • Naoto Urusihara • Koji Fukumoto • Go Miyano • Hiroshi Nouso • Keiichi Morita • Hiromu Miyake Masakatsu Kaneshiro
•
Accepted: 15 July 2014 / Published online: 23 July 2014 Ó Springer-Verlag Berlin Heidelberg 2014
Abstract Purpose The potential benefits of thoracoscopic repair (TR) of esophageal atresia and tracheoesophageal fistula (EA/TEF) in newborns are still unclear. Our aim was to define the criteria, perioperative outcome after undergoing TR versus open repair (OR) for EA/TEF. Patients and methods A retrospective chart review was conducted of 36 consecutive neonates who underwent EA/ TEF repair between 2001 and 2012 in Shizuoka Children’s Hospital. Patients in this study were birth weight [2,000 g, and did not have severe cardiac malformations or chromosomal aberrations. Of the 26 newborns who met the selection criteria, 11 patients underwent attempts at TR compared to 15 patients who underwent OR. All cases were followed 1 year after operation at least. Results All 11 TR were successfully completed. There were no significant differences between intra- and perioperative complications in the two groups. Intraoperative EtCO2 and arterial blood gases were not significantly different between the two groups. We did not found eating disorder, respiratory disorder, and failure of growth in all cases. Conclusion In our study, the thoracoscopic approach appeared to be favorable and safe for EA/TEF repair in carefully selected patients. Keywords Esophageal atresia Tracheoesophageal fistula Thoracoscopy One-lung ventilation
M. Yamoto (&) N. Urusihara K. Fukumoto G. Miyano H. Nouso K. Morita H. Miyake M. Kaneshiro Department of Pediatric Surgery, Shizuoka Children’s Hospital, 860 Urushiyama, Aoi-ku, Shizuoka 420-8660, Japan e-mail: [email protected]
Introduction Esophageal atresia (EA) with or without a tracheoesophageal fistula (TEF) is a rare congenital malformation occurring in 1 of 5,000 newborns [1]. The survival rate of patients with EA has increased significantly during the last few decades [2, 3], and has exceeded 90 % in recent years [2]. Most patients without severe associated malformations survive. The high mortality of very low birth weight patients with TEF and patients with severe cardiac malformations has also decreased significantly [4]. Conventionally, open, right-sided, muscle-sparing thoracotomy (open repair, OR) is the standard approach for repair of EA/TEF in many surgical centers [3, 5, 6]. In 1999, the first thoracoscopic repair (TR) of isolated EA was performed in a 2-month-old infant [6]. One year later, the first total TR of an atresia with distal fistula was performed in a newborn [7]. These milestones allowed for more widespread adoption of this technique, and an increasing number of pediatric surgical units are now performing minimally invasive EA/TEF repair [1]. Advances in pediatric thoracoscopy allow performance of sophisticated procedures, even in the confined anatomic spaces afforded by neonates [3]. Recently, advances and excellent feasibility of thoracoscopic EA/TEF repair have been reported with increasing frequency [1, 3, 8–10]. However, the rate of intra- and postoperative complications seems to be higher compared with the open approach [9– 11]. In our department, a TR for uncomplicated EA/TEF was first performed in 2008. The choice of thoracoscopy is based on precise selection criteria. The aim of this study was to evaluate the safety and efficacy of TR of EA/TEF using our selective approach. The endpoints assessed were intra- and perioperative complications.
123
884
Patients and methods The study was approved by the local Institutional Ethics Committee (No. 2013098). A retrospective chart review of all consecutive patients undergoing EA/TEF repair between January 2001 and March 2012, which included 36 newborns with EA/TEF who were admitted to Shizuoka Children’s Hospital, was conducted. All patients underwent standardized preoperative assessment, including full physical examination, chest radiography, echocardiography and abdominal ultrasound. The criteria of thoracoscopic repair at our institution were birth weight [2,000 g and the absence of severe cardiac malformations (e.g., single ventricle) and chromosomal aberrations (e.g., trisomy 18 and trisomy 13). Twenty-six of the 36 patients met these criteria and were included in this study. The reasons for exclusion of the remaining ten patients were birth weight \2,000 g in five cases, severe cardiac malformations in three, and severe chromosomal aberrations in two. Of the 26 newborns, 11 patients (7 boys and 4 girls) underwent attempts at TR, compared to 15 (11 boys and 4 girls) patients who underwent OR. OR procedures were performed by different residents and fellows under the supervision of attending surgeons. TRs were performed by a single surgeon. The data collected included the newborn’s age and weight at the time of the operation, associated malformations, esophageal gap, anesthesia time, operative time, blood loss, rate of conversion to OR, intraoperative EtCO2, arterial blood gases, days to extubation, time to first feeding, total duration of hospitalization, and number of patients with leak and/or stricture formation, fundoplication, recurrent fistula, phrenic nerve paralysis and tracheomalacia. Intraoperatively, blood gases were measured at the time of creation of the pneumothorax in the thoracoscopic procedure, and at pulmonary exclusion in patients undergoing thoracotomy. Intraoperatively, all children were continuously monitored for end-tidal pCO2 (EtCO2) values. Postoperative monitoring in all patients was carried out in the pediatric intensive care unit. During surgery and as long as indicated in the pediatric intensive care unit, all patients were on pressure-controlled ventilation. Weaning and extubation depended on the postoperative course of individual patients, with the decision being made jointly by both the pediatric intensive care physician and the pediatric surgeon. All patients in the TR and OR group were followed 1 year later after operation at least.
Pediatr Surg Int (2014) 30:883–887
tracheal intubation was performed with a 3.0-mm tracheal tube. Flexible bronchoscopy was performed, and the fistula was cannulated with the Fogarty catheter to block the fistula. After occlusion of the fistula, a tracheal tube was inserted in the left main bronchus and one-lung ventilation was performed. Tracheal intubation with an attempt at onelung ventilation was performed in eight TR cases and nine OR cases. Occlusion of the fistula was performed in nine TR cases and three OR cases. Thoracoscopy The patient was then placed in a semiprone position with the right side elevated by 45°. A 5-mm trocar was inserted approximately 1 cm below the angle of the scapula. Insufflation of CO2 was initiated at the rate of 0.5 L/min, creating an intrathoracic pressure of 4–8 mmHg, which was then decreased following respiratory adaptation of the patient. Thereafter, a 5-mm thoracoscope was used. Two additional 3-mm working ports were placed under direct vision in the 6th or 7th intercostal space in the mid-axillary line and the 3rd intercostal space in the anterior axillary line. A fourth was placed either higher or lower in the thoracic cavity to help retract the lung, but this was not necessary in the majority of our cases. First, the mediastinal pleura was opened longitudinally just beneath the azygos vein and dissection was commenced. To prevent recurrence of the fistula, we did not divide the azygos vein. The azygos vein was not ligated and transected. The lower part of the esophagus was mobilized for only a very short distance. A nasogastric tube was then inserted by the anesthetist to clearly identify and mobilize the proximal esophagus. The distal fistula was closed by transfixing sutures using 4/0 PDS (polydioxanone, Ethicon, Inc., Somervillle, NJ, USA), and the proximal esophagus was then opened. Following partial transection of the distal fistula, the first anastomotic suture was placed while avoiding excessive tension. Transection of the distal fistula was completed, along with completion of the first anastomotic suture. Semicircular sutures were placed for the anastomosis, and the NG tube was advanced through the distal esophagus into the stomach under direct vision. The anastomosis was then completed circumferentially using 6–10 interrupted 5/0 Vicryl (polyglactin, Ethicon, Inc.) sutures. A chest drain was inserted under direct vision through the trocar site, with the tip being placed beside the anastomosis.
Surgical technique Thoracotomy Anesthesia Under general anesthesia, a 4-Fr Fogarty catheter was inserted via a nostril into the trachea, following which
123
For children undergoing open surgery, a lateral thoracotomy with partial dissection of the latissimus dorsi as well as the serratus muscle was performed. An intra- or extra-
Pediatr Surg Int (2014) 30:883–887
pleural approach to the mediastinum was adopted. The azygos vein was ligated and transected in all cases. After identification and mobilization of the proximal esophagus, the anastomosis was performed as in the thoracoscopic approach. Closure of the thoracotomy was carried out in anatomical layers using interrupted 5/0 or 6/0 Vicryl sutures. A transanastomotic tube and chest drain was used in all cases. Postoperative course Sedation and analgesia without muscle relaxants were administered to all patients until 3 days after surgery. Enteral feeding with breast milk or regular milk was initiated on postoperative day 5. Oral feeding was started, when a contrast study was done to prove the patent anastomosis. The nasogastric tube was removed, when an oral intake became full feeding. One month postoperatively, all patients underwent anastomotic balloon dilatation of the esophagus to 6–8 mm. The need for at least two dilatations was defined as an anastomotic stricture.
885 Table 1 Patient demographic data Variable
TR group (n = 11)
OR group (n = 15)
P value
Gender (male, %)
63.6
73.3
0.68
Mean gestational age (weeks)
38.6 (36–40)
38.5 (37–40)
0.57
Median birth weight (g)
2,649 (2,100–3,104)
2,712 (2,210–3,064)
0.69
Median age at operation (days)
1.9 (0–7)
3.7 (0–10)
0.01*
Esophageal gap (distance of vertebrae)
1.4 (0–2.5)
0.6 (0–2)
0.03*
Associated anomalies (%)
54.5
46.7
0.52
TR thoracoscopic repair, OR open repair * P \ 0.05
Table 2 Comparison of clinical data (thoracoscopic repair versus open repair) Variable
TR group (n = 11)
OR group (n = 15)
P value
Anesthesia time
288.5 (252–360)
241.1 (200–290)
0.01*
One-lung ventilation (%)
72.7
60
0.68
Occlusion of TEF (%)
81.8
20
0.002*
Operative time (min)
174.5 (135–260)
155.3 (120–190)
0.07
Bleeding (g)
1 (0–10)
12 (5–20)
0.01*
Intraoperative complications (%)
0
0
NA
Statistical evaluation All results are expressed as median (range). The Wilcoxon rank sum test was used to compare continuous variables between the two groups. Fisher’s test for discrete variables was used to compare the two groups. For all analyses, a P value of \0.05 was considered significant. Data analysis was done with the JMPÒ 9 statistical software (SAS Institute, Cary, NC, USA).
Results Repair was successfully completed thoracoscopically in all 11 patients in whom it was attempted. Mean gestational age was 38.6 weeks (range 36–40) versus 38.5 weeks (range 37–40); age at operation was 1.9 days (range 0–7) versus 3.7 days (range 0–10); and weight at the time of surgery was 2.6 kg (range 2.1–3.1) versus 2.7 kg (range 2.2–3.1) in the TR and OR groups, respectively. At least one major associated anomaly was present in 54.5 % of patients in the TR group versus 46.7 % of patients in the OR group (Table 1). Intraoperative one-lung ventilation was performed in 73 % of the TR group patients versus 60 % of OR group patients; occlusion of the tracheoesophageal fistula with the Fogarty catheter was done in 81.8 % of TR group patients versus 20 % of the OR patients, which was a significant difference (P \ 0.005). Overall, the mean operative time was 174.5 min (range 135–260) in the TR group, versus 155.3 min (range 120–190) in the OR group; the mean anesthesia time was
TR thoracoscopic repair, OR open repair, TEF tracheoesophageal fistula * P \ 0.05
288.5 min (range 252–360) versus 241.1 min (range 200–290) in the TR and OR groups, respectively, the difference between them being significantly different (P \ 0.005); mean blood loss was 1 cc (range 0–10) versus 12 cc (range 5–20) in the TR and OR groups, respectively, the difference between the two being significant (P \ 0.005); there were no intraoperative complications or deaths in either group (Table 2). The stricture rate was 27 % in the TR group versus 33 % in the OR group. The anastomotic leak rate was 9 % in the TR group versus 13 % in the OR group. All leaks resolved spontaneously. Eighteen percent versus 20 % patients in the TR and OR groups, respectively, required fundoplication for severe gastro-esophageal reflux. Three patients in the OR group were diagnosed with recurrence of the tracheoesophageal fistula, which required reoperation. The mean time to extubation was 3.5 days (range 2–5) versus 5.6 days (range
123
886
3–15) in the TR and OR groups, respectively, which was a significant difference (P \ 0.005); the mean time to first oral feeding was 8.9 days (range 7–11) versus 11.3 days (range 7–20) in the TR and OR groups; and the mean duration of total hospitalization was 56.9 days (range 24–210) versus 67.7 days (range 23–271) in the TR and OR groups, respectively. The growth state (body weight, height) of all patients were within ±1 SD, 1 year after operation. Eating disorders and respiratory disorders were not found in all cases (Table 3). The median intraoperative values of pCO2, pO2, pH, BE, EtCO2max and EtCO2min were not significantly different between the two groups (Table 4).
Discussion Thoracotomy is considered standard surgery for repair of esophageal atresia. With advances in pediatric endoscopic surgery and anesthesia, the indications for a thoracoscopic approach are increasing. TR offers good surgical outcomes and cosmetic advantages. Several reports have assessed the safety and effectiveness of TR [9, 12–14]. The main operative advantage of TR is dissection of the posterior mediastinal structures under excellent visualization. Furthermore, TR enables avoidance of scoliosis, which occurs as a consequence of OR. Moreover, trauma due to incision of the parietal pleura with costal separation results in more pain, stress and morbidity [13]. Despite all its advantages, minimally invasive repair of EA/TEF remains a technically challenging operation, with suturing of the anastomosis being the major hurdle. It is commonly accepted that surgical expertise and advanced thoracoscopic skills are required for successful completion of this operation [1, 10, 13]. The most difficult part of the operation is the anastomosis, which requires a certain amount of expertise in endoscopic suturing. All the knots were tied intracorporeally, which we believe is a more precise method than the extracorporeal technique, especially in newborns. The azygos vein was never divided in the TR group. Patkowsk et al. postulated that preservation of the azygos vein prevents early postoperative edema of the esophageal anastomosis by maintaining venous drainage, which may thus serve as an additional protective factor against anastomotic leaks, since the azygos vein separates the anastomosis from the site of fistula ligation, potentially preventing recurrent fistula formation [8]. In our study, recurrent fistulas occurred in three patients in the OR group in whom the azygos vein was divided. Conversely, we observed no fistula recurrence in the TR group, probably because the azygos vein was intact. Besides the surgeon’s expertise, we strongly believe that meticulous patient selection contributes to the success of
123
Pediatr Surg Int (2014) 30:883–887 Table 3 Comparison of postoperative clinical data (thoracoscopic repair versus open repair) Variable
TR group (n = 11)
OR group (n = 15)
P value
Stricture (%)
27.3
33.3
0.76
Anastomotic leakage (%) Fundoplication for GER (%)
18.2 18.1
20 20
0.91 0.91
Postoperative complication
Recurrent fistula (%)
0
20
0.23
Phrenic nerve paralysis (%)
0
6.7
0.57
Tracheomalacia (%)
9.1
6.7
0.81
Time to extubation (days)
3.5 (2–5)
5.6 (3–10)
0.01*
Time to first oral feeding (days)
8.9 (7–13)
11.3 (5–15)
0.13
Total of duration of hospitalization (days)
56.9 (24–210)
67.7 (39–271)
0.08
NA
Follow up data 1 year after operation Eating disorder (%)
0
0
Respiratory disorder (%)
0
0
NA
Failure of growth (%)
0
0
NA
TR thoracoscopic repair, OR open repair, GER gastro-esophageal reflux * P \ 0.05
Table 4 Comparisons of intraoperative EtCO2 and arterial blood gases (thoracoscopic repair versus open repair) Variable
TR group (n = 11)
OR group (n = 15)
P value
EtCO2max
26.4 (18 to 31)
27.5 (24 to 30)
EtCO2min
36 (27 to 43)
37.4 (32 to 48)
0.67
7.35 (7.15 to 7.45)
0.31
0.77
pH
7.31 (7.22 to 7.38)
pCO2
43.8 (33 to 47)
47.6 (34 to 55)
0.85
pO2
93.8 (46 to 128)
104.9 (48 to 188)
0.2
BE
-2.9 (-6.3 to 0.1)
-0.2 (-4.8 to 4.7)
0.06
TR thoracoscopic repair, OR open repair
this minimally invasive procedure. Rothenberg et al. [12] reported that severe hemodynamic instability requiring significant pressor support, and significant prematurity (birth weight \1,500 g) are absolute contraindications to a thoracoscopic approach. Relative contraindications include significant congenital cardiac defects, smaller size (1,500–2,000 g), or significant abdominal distension. The criteria of thoracoscopic repair at our institution were birth weight [2,000 g and the absence of severe cardiac malformations (e.g., single ventricle) and chromosomal aberrations (e.g., trisomy 18 and trisomy 13). We suggest the use of the aforementioned criteria in order to provide the best possible treatment to EA/TEF patients and to ensure a postoperative outcome at least similar to that following
Pediatr Surg Int (2014) 30:883–887
open operation. In our series, no deaths were observed after TR. We believe that our principle of careful patient selection contributed to this favorable outcome. Relatively few studies reporting cardiorespiratory consequences of thoracoscopy in neonates have been published [10, 15], and those that have been published relied on retrospective review. A randomized study showed that thoracoscopic repair of congenital diaphragmatic hernias is associated with profound and prolonged hypercapnia and acidosis compared with open surgery. In EA/TEF cases, TR is reportedly not associated with hypercapnia and acidosis [16]. This is now the subject of further investigation. In our experience, intraoperative values of blood gases, EtCO2max, and EtCO2min were not significantly different in the OR and TR groups. Borruto et al. [17] analyzed the outcomes that are universally considered to be indicators of the effectiveness of the thoracoscopic EA/TEF repair, namely, the rates of complications, conversion to OR, anastomotic leaks and strictures. In their meta-analysis, they demonstrated statistically insignificant differences in all the parameters considered. In our series, we found statistically insignificant differences in intra- and postoperative complications in the two groups. We, thus, demonstrated that TR of EA/ TEF provides similar results as OR. More studies will be necessary to establish the absolute efficacy and safety of the minimally invasive technique.
Conclusion In our study, the thoracoscopic approach appeared to be favorable and safe for EA/TEF repair in carefully selected patients. Conflict of interest The authors declare that they have no conflicts of interest.
References
887 2. Goyal A, Jones MO, Couriel JM, Losty PD (2006) Oesophageal atresia and tracheo-oesophageal fistula. Arch Dis Child Fetal Neonatal Ed 91(5):F381–F384 3. Rothenberg SS (2002) Thoracoscopic repair of tracheoesophageal fistula in newborns. J Pediatr Surg 37(6):869–872 4. Rintala RJ, Sistonen S, Pakarinen MP (2009) Outcome of esophageal atresia beyond childhood. Semin Pediatr Surg 18(1):50–56 5. Mortell AE, Azizkhan RG (2009) Esophageal atresia repair with thoracotomy: the Cincinnati contemporary experience. Semin Pediatr Surg 18(1):12–19 6. Lobe TE, Rothenberg SS, Waldschmidt J (1999) Thoracoscopic repair of esophageal atresia in an infant: a surgical first. Pediatr Endosurg Innov Tech 3:141–148 7. Rothenberg SS (2000) Thoracoscopic repair of tracheoesophageal fistula in a newborn infant. Pediatr Endosurg Innov Tech 4:289–294 8. van der Zee DC, Bax KN (2007) Thoracoscopic treatment of esophageal atresia with distal fistula and of tracheomalacia. Semin Pediatr Surg 16(4):224–230 9. MacKinlay GA (2009) Esophageal atresia surgery in the 21st century. Semin Pediatr Surg 18(1):20–22 10. Patkowsk D, Rysiakiewicz K, Jaworski W et al (2009) Thoracoscopic repair of tracheoesophageal fistula and esophageal atresia. J Laparoendosc Adv Surg Tech A 19(Suppl 1):S19–S22 11. van der Zee DC, Vieirra-Travassos D, de Jong JR, Tytgat SH (2008) A novel technique for risk calculation of anastomotic leakage after thoracoscopic repair for esophageal atresia with distal fistula. World J Surg 32(7):1396–1399 12. Rothenberg SS (2005) Thoracoscopic repair of esophageal atresia and tracheo-esophageal fistula. Semin Pediatr Surg 14(1):2–7 13. Holcomb GW 3rd, Rothenberg SS, Bax KM et al (2005) Thoracoscopic repair of esophageal atresia and tracheoesophageal fistula: a multi-institutional analysis. Ann Surg 242(3):422–430 14. Bax KM, van Der Zee DC (2002) Feasibility of thoracoscopic repair of esophageal atresia with distal fistula. J Pediatr Surg 37(2):192–196 15. Krosnar S, Baxter A (2005) Thoracoscopic repair of esophageal atresia with tracheoesophageal fistula: anesthetic and intensive care management of a series of eight neonates. Paediatr Anaesth 15:541–546 16. Bishay M, Giacomello L, Retrosi G et al (2013) Hypercapnia and acidosis during open and thoracoscopic repair of congenital diaphragmatic hernia and esophageal atresia: results of a pilot randomized controlled trial. Ann Surg 258(6):895–900 17. Borruto FA, Impellizzeri P, Montalto AS et al (2012) Thoracoscopy versus thoracotomy for esophageal atresia and tracheoesophageal fistula repair: review of the literature and metaanalysis. Eur J Pediatr Surg 22(6):415–419
1. Rothenberg SS (2012) Thoracoscopic repair of esophageal atresia and tracheo-esophageal fistula in neonates: evolution of a technique. J Laparoendosc Adv Surg Tech A 22(2):195–199
123
ORIGINAL ARTICLE
Thoracoscopic versus open repair of esophageal atresia with tracheoesophageal fistula at a single institution Masaya Yamoto • Naoto Urusihara • Koji Fukumoto • Go Miyano • Hiroshi Nouso • Keiichi Morita • Hiromu Miyake Masakatsu Kaneshiro
•
Accepted: 15 July 2014 / Published online: 23 July 2014 Ó Springer-Verlag Berlin Heidelberg 2014
Abstract Purpose The potential benefits of thoracoscopic repair (TR) of esophageal atresia and tracheoesophageal fistula (EA/TEF) in newborns are still unclear. Our aim was to define the criteria, perioperative outcome after undergoing TR versus open repair (OR) for EA/TEF. Patients and methods A retrospective chart review was conducted of 36 consecutive neonates who underwent EA/ TEF repair between 2001 and 2012 in Shizuoka Children’s Hospital. Patients in this study were birth weight [2,000 g, and did not have severe cardiac malformations or chromosomal aberrations. Of the 26 newborns who met the selection criteria, 11 patients underwent attempts at TR compared to 15 patients who underwent OR. All cases were followed 1 year after operation at least. Results All 11 TR were successfully completed. There were no significant differences between intra- and perioperative complications in the two groups. Intraoperative EtCO2 and arterial blood gases were not significantly different between the two groups. We did not found eating disorder, respiratory disorder, and failure of growth in all cases. Conclusion In our study, the thoracoscopic approach appeared to be favorable and safe for EA/TEF repair in carefully selected patients. Keywords Esophageal atresia Tracheoesophageal fistula Thoracoscopy One-lung ventilation
M. Yamoto (&) N. Urusihara K. Fukumoto G. Miyano H. Nouso K. Morita H. Miyake M. Kaneshiro Department of Pediatric Surgery, Shizuoka Children’s Hospital, 860 Urushiyama, Aoi-ku, Shizuoka 420-8660, Japan e-mail: [email protected]
Introduction Esophageal atresia (EA) with or without a tracheoesophageal fistula (TEF) is a rare congenital malformation occurring in 1 of 5,000 newborns [1]. The survival rate of patients with EA has increased significantly during the last few decades [2, 3], and has exceeded 90 % in recent years [2]. Most patients without severe associated malformations survive. The high mortality of very low birth weight patients with TEF and patients with severe cardiac malformations has also decreased significantly [4]. Conventionally, open, right-sided, muscle-sparing thoracotomy (open repair, OR) is the standard approach for repair of EA/TEF in many surgical centers [3, 5, 6]. In 1999, the first thoracoscopic repair (TR) of isolated EA was performed in a 2-month-old infant [6]. One year later, the first total TR of an atresia with distal fistula was performed in a newborn [7]. These milestones allowed for more widespread adoption of this technique, and an increasing number of pediatric surgical units are now performing minimally invasive EA/TEF repair [1]. Advances in pediatric thoracoscopy allow performance of sophisticated procedures, even in the confined anatomic spaces afforded by neonates [3]. Recently, advances and excellent feasibility of thoracoscopic EA/TEF repair have been reported with increasing frequency [1, 3, 8–10]. However, the rate of intra- and postoperative complications seems to be higher compared with the open approach [9– 11]. In our department, a TR for uncomplicated EA/TEF was first performed in 2008. The choice of thoracoscopy is based on precise selection criteria. The aim of this study was to evaluate the safety and efficacy of TR of EA/TEF using our selective approach. The endpoints assessed were intra- and perioperative complications.
123
884
Patients and methods The study was approved by the local Institutional Ethics Committee (No. 2013098). A retrospective chart review of all consecutive patients undergoing EA/TEF repair between January 2001 and March 2012, which included 36 newborns with EA/TEF who were admitted to Shizuoka Children’s Hospital, was conducted. All patients underwent standardized preoperative assessment, including full physical examination, chest radiography, echocardiography and abdominal ultrasound. The criteria of thoracoscopic repair at our institution were birth weight [2,000 g and the absence of severe cardiac malformations (e.g., single ventricle) and chromosomal aberrations (e.g., trisomy 18 and trisomy 13). Twenty-six of the 36 patients met these criteria and were included in this study. The reasons for exclusion of the remaining ten patients were birth weight \2,000 g in five cases, severe cardiac malformations in three, and severe chromosomal aberrations in two. Of the 26 newborns, 11 patients (7 boys and 4 girls) underwent attempts at TR, compared to 15 (11 boys and 4 girls) patients who underwent OR. OR procedures were performed by different residents and fellows under the supervision of attending surgeons. TRs were performed by a single surgeon. The data collected included the newborn’s age and weight at the time of the operation, associated malformations, esophageal gap, anesthesia time, operative time, blood loss, rate of conversion to OR, intraoperative EtCO2, arterial blood gases, days to extubation, time to first feeding, total duration of hospitalization, and number of patients with leak and/or stricture formation, fundoplication, recurrent fistula, phrenic nerve paralysis and tracheomalacia. Intraoperatively, blood gases were measured at the time of creation of the pneumothorax in the thoracoscopic procedure, and at pulmonary exclusion in patients undergoing thoracotomy. Intraoperatively, all children were continuously monitored for end-tidal pCO2 (EtCO2) values. Postoperative monitoring in all patients was carried out in the pediatric intensive care unit. During surgery and as long as indicated in the pediatric intensive care unit, all patients were on pressure-controlled ventilation. Weaning and extubation depended on the postoperative course of individual patients, with the decision being made jointly by both the pediatric intensive care physician and the pediatric surgeon. All patients in the TR and OR group were followed 1 year later after operation at least.
Pediatr Surg Int (2014) 30:883–887
tracheal intubation was performed with a 3.0-mm tracheal tube. Flexible bronchoscopy was performed, and the fistula was cannulated with the Fogarty catheter to block the fistula. After occlusion of the fistula, a tracheal tube was inserted in the left main bronchus and one-lung ventilation was performed. Tracheal intubation with an attempt at onelung ventilation was performed in eight TR cases and nine OR cases. Occlusion of the fistula was performed in nine TR cases and three OR cases. Thoracoscopy The patient was then placed in a semiprone position with the right side elevated by 45°. A 5-mm trocar was inserted approximately 1 cm below the angle of the scapula. Insufflation of CO2 was initiated at the rate of 0.5 L/min, creating an intrathoracic pressure of 4–8 mmHg, which was then decreased following respiratory adaptation of the patient. Thereafter, a 5-mm thoracoscope was used. Two additional 3-mm working ports were placed under direct vision in the 6th or 7th intercostal space in the mid-axillary line and the 3rd intercostal space in the anterior axillary line. A fourth was placed either higher or lower in the thoracic cavity to help retract the lung, but this was not necessary in the majority of our cases. First, the mediastinal pleura was opened longitudinally just beneath the azygos vein and dissection was commenced. To prevent recurrence of the fistula, we did not divide the azygos vein. The azygos vein was not ligated and transected. The lower part of the esophagus was mobilized for only a very short distance. A nasogastric tube was then inserted by the anesthetist to clearly identify and mobilize the proximal esophagus. The distal fistula was closed by transfixing sutures using 4/0 PDS (polydioxanone, Ethicon, Inc., Somervillle, NJ, USA), and the proximal esophagus was then opened. Following partial transection of the distal fistula, the first anastomotic suture was placed while avoiding excessive tension. Transection of the distal fistula was completed, along with completion of the first anastomotic suture. Semicircular sutures were placed for the anastomosis, and the NG tube was advanced through the distal esophagus into the stomach under direct vision. The anastomosis was then completed circumferentially using 6–10 interrupted 5/0 Vicryl (polyglactin, Ethicon, Inc.) sutures. A chest drain was inserted under direct vision through the trocar site, with the tip being placed beside the anastomosis.
Surgical technique Thoracotomy Anesthesia Under general anesthesia, a 4-Fr Fogarty catheter was inserted via a nostril into the trachea, following which
123
For children undergoing open surgery, a lateral thoracotomy with partial dissection of the latissimus dorsi as well as the serratus muscle was performed. An intra- or extra-
Pediatr Surg Int (2014) 30:883–887
pleural approach to the mediastinum was adopted. The azygos vein was ligated and transected in all cases. After identification and mobilization of the proximal esophagus, the anastomosis was performed as in the thoracoscopic approach. Closure of the thoracotomy was carried out in anatomical layers using interrupted 5/0 or 6/0 Vicryl sutures. A transanastomotic tube and chest drain was used in all cases. Postoperative course Sedation and analgesia without muscle relaxants were administered to all patients until 3 days after surgery. Enteral feeding with breast milk or regular milk was initiated on postoperative day 5. Oral feeding was started, when a contrast study was done to prove the patent anastomosis. The nasogastric tube was removed, when an oral intake became full feeding. One month postoperatively, all patients underwent anastomotic balloon dilatation of the esophagus to 6–8 mm. The need for at least two dilatations was defined as an anastomotic stricture.
885 Table 1 Patient demographic data Variable
TR group (n = 11)
OR group (n = 15)
P value
Gender (male, %)
63.6
73.3
0.68
Mean gestational age (weeks)
38.6 (36–40)
38.5 (37–40)
0.57
Median birth weight (g)
2,649 (2,100–3,104)
2,712 (2,210–3,064)
0.69
Median age at operation (days)
1.9 (0–7)
3.7 (0–10)
0.01*
Esophageal gap (distance of vertebrae)
1.4 (0–2.5)
0.6 (0–2)
0.03*
Associated anomalies (%)
54.5
46.7
0.52
TR thoracoscopic repair, OR open repair * P \ 0.05
Table 2 Comparison of clinical data (thoracoscopic repair versus open repair) Variable
TR group (n = 11)
OR group (n = 15)
P value
Anesthesia time
288.5 (252–360)
241.1 (200–290)
0.01*
One-lung ventilation (%)
72.7
60
0.68
Occlusion of TEF (%)
81.8
20
0.002*
Operative time (min)
174.5 (135–260)
155.3 (120–190)
0.07
Bleeding (g)
1 (0–10)
12 (5–20)
0.01*
Intraoperative complications (%)
0
0
NA
Statistical evaluation All results are expressed as median (range). The Wilcoxon rank sum test was used to compare continuous variables between the two groups. Fisher’s test for discrete variables was used to compare the two groups. For all analyses, a P value of \0.05 was considered significant. Data analysis was done with the JMPÒ 9 statistical software (SAS Institute, Cary, NC, USA).
Results Repair was successfully completed thoracoscopically in all 11 patients in whom it was attempted. Mean gestational age was 38.6 weeks (range 36–40) versus 38.5 weeks (range 37–40); age at operation was 1.9 days (range 0–7) versus 3.7 days (range 0–10); and weight at the time of surgery was 2.6 kg (range 2.1–3.1) versus 2.7 kg (range 2.2–3.1) in the TR and OR groups, respectively. At least one major associated anomaly was present in 54.5 % of patients in the TR group versus 46.7 % of patients in the OR group (Table 1). Intraoperative one-lung ventilation was performed in 73 % of the TR group patients versus 60 % of OR group patients; occlusion of the tracheoesophageal fistula with the Fogarty catheter was done in 81.8 % of TR group patients versus 20 % of the OR patients, which was a significant difference (P \ 0.005). Overall, the mean operative time was 174.5 min (range 135–260) in the TR group, versus 155.3 min (range 120–190) in the OR group; the mean anesthesia time was
TR thoracoscopic repair, OR open repair, TEF tracheoesophageal fistula * P \ 0.05
288.5 min (range 252–360) versus 241.1 min (range 200–290) in the TR and OR groups, respectively, the difference between them being significantly different (P \ 0.005); mean blood loss was 1 cc (range 0–10) versus 12 cc (range 5–20) in the TR and OR groups, respectively, the difference between the two being significant (P \ 0.005); there were no intraoperative complications or deaths in either group (Table 2). The stricture rate was 27 % in the TR group versus 33 % in the OR group. The anastomotic leak rate was 9 % in the TR group versus 13 % in the OR group. All leaks resolved spontaneously. Eighteen percent versus 20 % patients in the TR and OR groups, respectively, required fundoplication for severe gastro-esophageal reflux. Three patients in the OR group were diagnosed with recurrence of the tracheoesophageal fistula, which required reoperation. The mean time to extubation was 3.5 days (range 2–5) versus 5.6 days (range
123
886
3–15) in the TR and OR groups, respectively, which was a significant difference (P \ 0.005); the mean time to first oral feeding was 8.9 days (range 7–11) versus 11.3 days (range 7–20) in the TR and OR groups; and the mean duration of total hospitalization was 56.9 days (range 24–210) versus 67.7 days (range 23–271) in the TR and OR groups, respectively. The growth state (body weight, height) of all patients were within ±1 SD, 1 year after operation. Eating disorders and respiratory disorders were not found in all cases (Table 3). The median intraoperative values of pCO2, pO2, pH, BE, EtCO2max and EtCO2min were not significantly different between the two groups (Table 4).
Discussion Thoracotomy is considered standard surgery for repair of esophageal atresia. With advances in pediatric endoscopic surgery and anesthesia, the indications for a thoracoscopic approach are increasing. TR offers good surgical outcomes and cosmetic advantages. Several reports have assessed the safety and effectiveness of TR [9, 12–14]. The main operative advantage of TR is dissection of the posterior mediastinal structures under excellent visualization. Furthermore, TR enables avoidance of scoliosis, which occurs as a consequence of OR. Moreover, trauma due to incision of the parietal pleura with costal separation results in more pain, stress and morbidity [13]. Despite all its advantages, minimally invasive repair of EA/TEF remains a technically challenging operation, with suturing of the anastomosis being the major hurdle. It is commonly accepted that surgical expertise and advanced thoracoscopic skills are required for successful completion of this operation [1, 10, 13]. The most difficult part of the operation is the anastomosis, which requires a certain amount of expertise in endoscopic suturing. All the knots were tied intracorporeally, which we believe is a more precise method than the extracorporeal technique, especially in newborns. The azygos vein was never divided in the TR group. Patkowsk et al. postulated that preservation of the azygos vein prevents early postoperative edema of the esophageal anastomosis by maintaining venous drainage, which may thus serve as an additional protective factor against anastomotic leaks, since the azygos vein separates the anastomosis from the site of fistula ligation, potentially preventing recurrent fistula formation [8]. In our study, recurrent fistulas occurred in three patients in the OR group in whom the azygos vein was divided. Conversely, we observed no fistula recurrence in the TR group, probably because the azygos vein was intact. Besides the surgeon’s expertise, we strongly believe that meticulous patient selection contributes to the success of
123
Pediatr Surg Int (2014) 30:883–887 Table 3 Comparison of postoperative clinical data (thoracoscopic repair versus open repair) Variable
TR group (n = 11)
OR group (n = 15)
P value
Stricture (%)
27.3
33.3
0.76
Anastomotic leakage (%) Fundoplication for GER (%)
18.2 18.1
20 20
0.91 0.91
Postoperative complication
Recurrent fistula (%)
0
20
0.23
Phrenic nerve paralysis (%)
0
6.7
0.57
Tracheomalacia (%)
9.1
6.7
0.81
Time to extubation (days)
3.5 (2–5)
5.6 (3–10)
0.01*
Time to first oral feeding (days)
8.9 (7–13)
11.3 (5–15)
0.13
Total of duration of hospitalization (days)
56.9 (24–210)
67.7 (39–271)
0.08
NA
Follow up data 1 year after operation Eating disorder (%)
0
0
Respiratory disorder (%)
0
0
NA
Failure of growth (%)
0
0
NA
TR thoracoscopic repair, OR open repair, GER gastro-esophageal reflux * P \ 0.05
Table 4 Comparisons of intraoperative EtCO2 and arterial blood gases (thoracoscopic repair versus open repair) Variable
TR group (n = 11)
OR group (n = 15)
P value
EtCO2max
26.4 (18 to 31)
27.5 (24 to 30)
EtCO2min
36 (27 to 43)
37.4 (32 to 48)
0.67
7.35 (7.15 to 7.45)
0.31
0.77
pH
7.31 (7.22 to 7.38)
pCO2
43.8 (33 to 47)
47.6 (34 to 55)
0.85
pO2
93.8 (46 to 128)
104.9 (48 to 188)
0.2
BE
-2.9 (-6.3 to 0.1)
-0.2 (-4.8 to 4.7)
0.06
TR thoracoscopic repair, OR open repair
this minimally invasive procedure. Rothenberg et al. [12] reported that severe hemodynamic instability requiring significant pressor support, and significant prematurity (birth weight \1,500 g) are absolute contraindications to a thoracoscopic approach. Relative contraindications include significant congenital cardiac defects, smaller size (1,500–2,000 g), or significant abdominal distension. The criteria of thoracoscopic repair at our institution were birth weight [2,000 g and the absence of severe cardiac malformations (e.g., single ventricle) and chromosomal aberrations (e.g., trisomy 18 and trisomy 13). We suggest the use of the aforementioned criteria in order to provide the best possible treatment to EA/TEF patients and to ensure a postoperative outcome at least similar to that following
Pediatr Surg Int (2014) 30:883–887
open operation. In our series, no deaths were observed after TR. We believe that our principle of careful patient selection contributed to this favorable outcome. Relatively few studies reporting cardiorespiratory consequences of thoracoscopy in neonates have been published [10, 15], and those that have been published relied on retrospective review. A randomized study showed that thoracoscopic repair of congenital diaphragmatic hernias is associated with profound and prolonged hypercapnia and acidosis compared with open surgery. In EA/TEF cases, TR is reportedly not associated with hypercapnia and acidosis [16]. This is now the subject of further investigation. In our experience, intraoperative values of blood gases, EtCO2max, and EtCO2min were not significantly different in the OR and TR groups. Borruto et al. [17] analyzed the outcomes that are universally considered to be indicators of the effectiveness of the thoracoscopic EA/TEF repair, namely, the rates of complications, conversion to OR, anastomotic leaks and strictures. In their meta-analysis, they demonstrated statistically insignificant differences in all the parameters considered. In our series, we found statistically insignificant differences in intra- and postoperative complications in the two groups. We, thus, demonstrated that TR of EA/ TEF provides similar results as OR. More studies will be necessary to establish the absolute efficacy and safety of the minimally invasive technique.
Conclusion In our study, the thoracoscopic approach appeared to be favorable and safe for EA/TEF repair in carefully selected patients. Conflict of interest The authors declare that they have no conflicts of interest.
References
887 2. Goyal A, Jones MO, Couriel JM, Losty PD (2006) Oesophageal atresia and tracheo-oesophageal fistula. Arch Dis Child Fetal Neonatal Ed 91(5):F381–F384 3. Rothenberg SS (2002) Thoracoscopic repair of tracheoesophageal fistula in newborns. J Pediatr Surg 37(6):869–872 4. Rintala RJ, Sistonen S, Pakarinen MP (2009) Outcome of esophageal atresia beyond childhood. Semin Pediatr Surg 18(1):50–56 5. Mortell AE, Azizkhan RG (2009) Esophageal atresia repair with thoracotomy: the Cincinnati contemporary experience. Semin Pediatr Surg 18(1):12–19 6. Lobe TE, Rothenberg SS, Waldschmidt J (1999) Thoracoscopic repair of esophageal atresia in an infant: a surgical first. Pediatr Endosurg Innov Tech 3:141–148 7. Rothenberg SS (2000) Thoracoscopic repair of tracheoesophageal fistula in a newborn infant. Pediatr Endosurg Innov Tech 4:289–294 8. van der Zee DC, Bax KN (2007) Thoracoscopic treatment of esophageal atresia with distal fistula and of tracheomalacia. Semin Pediatr Surg 16(4):224–230 9. MacKinlay GA (2009) Esophageal atresia surgery in the 21st century. Semin Pediatr Surg 18(1):20–22 10. Patkowsk D, Rysiakiewicz K, Jaworski W et al (2009) Thoracoscopic repair of tracheoesophageal fistula and esophageal atresia. J Laparoendosc Adv Surg Tech A 19(Suppl 1):S19–S22 11. van der Zee DC, Vieirra-Travassos D, de Jong JR, Tytgat SH (2008) A novel technique for risk calculation of anastomotic leakage after thoracoscopic repair for esophageal atresia with distal fistula. World J Surg 32(7):1396–1399 12. Rothenberg SS (2005) Thoracoscopic repair of esophageal atresia and tracheo-esophageal fistula. Semin Pediatr Surg 14(1):2–7 13. Holcomb GW 3rd, Rothenberg SS, Bax KM et al (2005) Thoracoscopic repair of esophageal atresia and tracheoesophageal fistula: a multi-institutional analysis. Ann Surg 242(3):422–430 14. Bax KM, van Der Zee DC (2002) Feasibility of thoracoscopic repair of esophageal atresia with distal fistula. J Pediatr Surg 37(2):192–196 15. Krosnar S, Baxter A (2005) Thoracoscopic repair of esophageal atresia with tracheoesophageal fistula: anesthetic and intensive care management of a series of eight neonates. Paediatr Anaesth 15:541–546 16. Bishay M, Giacomello L, Retrosi G et al (2013) Hypercapnia and acidosis during open and thoracoscopic repair of congenital diaphragmatic hernia and esophageal atresia: results of a pilot randomized controlled trial. Ann Surg 258(6):895–900 17. Borruto FA, Impellizzeri P, Montalto AS et al (2012) Thoracoscopy versus thoracotomy for esophageal atresia and tracheoesophageal fistula repair: review of the literature and metaanalysis. Eur J Pediatr Surg 22(6):415–419
1. Rothenberg SS (2012) Thoracoscopic repair of esophageal atresia and tracheo-esophageal fistula in neonates: evolution of a technique. J Laparoendosc Adv Surg Tech A 22(2):195–199
123
