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Influence of thoracoscopic esophageal atresia repair on esophageal motor function and gastroesophageal reflux
Journal of Pediatric Surgery (2009) 44, 2282–2286
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Influence of thoracoscopic esophageal atresia repair on esophageal motor function and gastroesophageal reflux☆ Hisayoshi Kawaharaa,⁎, Hiroomi Okuyamaa , Yasuyuki Mitania , Motonari Nomuraa , Keisuke Nosea , Akihiro Yonedaa , Toshimichi Hasegawaa , Akio Kubotaa , Masahiro Fukuzawab a
Department of Pediatric Surgery, Osaka Medical Center and Research Institute for Maternal and Child Health, Osaka 594-1101, Japan b Department of Pediatric Surgery, Osaka University Graduate School of Medicine, Osaka, 565-0871, Japan Received 20 July 2009; accepted 31 July 2009
Key words: Esophageal atresia; Gastroesophageal reflux; Thoracoscopic repair; Videomanometry; 24-Hour esophageal pH monitoring
Abstract Background/Purpose: Thoracoscopic repair has recently been attempted in newborns with esophageal atresia (EA), but it remains unclear whether thoracoscopic dissection reduces pathological gastroesophageal reflux. We investigated the influence of a thoracoscopic approach on esophageal motor function in patients with EA. Methods: Clinical and gastrointestinal data of 10 patients with EA with open repair (group A) and 7 with thoracoscopic repair (group B) were analyzed retrospectively. Videomanometry was conducted to investigate esophageal motor patterns. Esophageal acid exposure was evaluated with 24-hour esophageal pH monitoring. Data are expressed as medians and ranges. Results: Contractions in the distal esophagus were conspicuously absent in 1 and 3 patients in groups A and B, respectively (P = .26). There were no significant differences in esophageal acid exposure (5.5% [0.7%-24.6%] vs 3.7% [0.3%-56.8%]; P = .71) or mean esophageal acid reflux time (0.5 minutes [0.11.4 minutes] vs 0.5 minutes [0.1-1.3 minutes]; P = .87) between the 2 groups. Fundoplication was conducted in 2 patients in each group (P = .60), all of whom had conspicuously absent distal esophageal contractions. Those contractions were preserved in the remaining patients with the exception of 1 group B patient. Conclusion: There are unlikely to be benefits from thoracoscopic repair of EA in terms of postoperative esophageal motor function. © 2009 Elsevier Inc. All rights reserved.
Presented at the 42nd Annual Meeting of the Pacific Association of Pediatric Surgeons, Hong Kong, China, May 10-14, 2009. ☆ Hisayoshi Kawahara, MD, was supported by the Sukoyaka Grant for Maternal and Child Health. ⁎ Corresponding author. Tel.: +81 725 56 1220; fax: +81 725 56 5682. E-mail address: [email protected] (H. Kawahara). 0022-3468/$ – see front matter © 2009 Elsevier Inc. All rights reserved. doi:10.1016/j.jpedsurg.2009.07.049
The emphasis of esophageal atresia (EA) in the modern era has changed to reducing morbidity and improving the quality of life after correction of EA [1]. Since the first introduction by Rothenberg et al, minimal invasive thoracoscopic repair of EA has been conducted in advanced pediatric endosurgical centers [2-5]. It is believed to be a superior cosmetic method with fewer sequelae, including
Thoracoscopic esophageal atresia repair and GER thoracic deformities and scoliosis. However, it remains to be proven whether thoracoscopic dissection has a less detrimental effect on esophageal motor function, which possibly contributes to preventing pathological gastroesophageal reflux (GER) occurring after repair of EA [5]. We aimed to investigate the influence of a thoracoscopic approach on esophageal motor function in patients with EA.
1. Materials and methods A retrospective review of 17 consecutive patients with Gross type C EA undergoing surgical correction with primary esophageal anastomosis in the period between December 2002 and April 2007 was performed. Data collected from the medical records included birth weight, gestational age, prenatal diagnosis, associated anomalies, operative procedure, postoperative leak, and stricture, as well as esophageal dilatation for anastomotic stricture if conducted. In addition, data obtained with videomanometry and 24-hour esophageal pH study were analyzed. These examinations were conducted as routine follow-up studies or as required by a patient's clinical condition. Ethical approval of these exminations was obtained from the
Table 1 Case
departmental committee, and informed consent was obtained from the patients' guardians. Administration of H2-receptor antagonists and fundoplication were also recorded if used.
1.1. Patients with EA The characteristics of each patient are detailed in Table 1. Of the 17 patients, 10 underwent open repair of EA (group A) and 7 thoracoscopic repair (group B). There was a significant difference in birth weight between groups A and B (2450 g [range, 1464-2908 g] vs 2814 g [range, 2460-3710 g]; P = .02) but no significant difference in gestational age (38 weeks [range, 33-41 weeks] vs 40 weeks [range, 37-41 weeks]; P = .15). The incidences of prenatal diagnosis (P = .33), association with other anomalies (P N .99), and gastrostomy (P = .30) were not significantly different between the 2 groups. The age at primary repair of EA was 2 days (range, 012 days) and 1 day (range, 1-3 days) in groups A and B, respectively (P = .37). The incidence of a minor leak was not significantly different between the 2 groups (P N .99), and only case 3 in group B showed fever and C-reactive protein (CRP) elevation. Two patients in group B, but none in group A, required esophageal balloon dilatation (P = .15).
Characteristics of each subject Birth weight (g)
Gestational age (wk)
Group A (open repair) 1 2908 41 2 2042 37 3 2626 37 4 1464 33 5 2580 38 6 2411 41 7 1610 34
8 9
2283
2170 2900
36 40
10 2488 39 Group B (thoracoscopic repair) 1 2814 41
Prenatal diagnosis
− + − − − − +
− +
Associated anomalies
CHARGE association, ECD Laryngeal atresia
Dudenal atresia, VSD Polydactyly, tracheomalacia VACTER association, tetralogy of fallot, duodenal atresia, severe scoliosis Agenesis of right kidney DORV, duodenal atresia, rectourethral fistula
Gastrostomy
2
2664
37
+
3 4 5 6 7
2460 3710 2604 2988 3046
40 41 41 38 38
− − + + +
Treacher Collins syndrome, micrognathia CHARGE association, VSD, cerebellar hypoplasia, hydronephrosis
Postoperative upper gastrointestinal Leak
Stricture
+
8 3 1 3 1 1 12
− Minor Minor − − − −
− − − − − − −
+
1 3
Minor −
− −
0
−
−
1
Minor
−
3
−
−
1 1 1 1 2
Minor − − − −
+ − − − +
+ + +
+ +
Age at primary repair (d)
+
Duodenal atresia, VSD Hydronephrosis Rectovestibular fistula
ECD, endocardial cushion defect; VSD, ventricular septal defect; DORV, double outlet right ventricle.
2284
1.2. Thoracoscopic repair of EA All thoracoscopic repair was conducted by one of the authors (HO) [6]. The patient was positioned prone with the right side slightly elevated. A 3-cm camera port was inserted into the sixth intercostal space. The pleural space was insufflated to a pressure of 3 to 4 mm Hg. Additional 3 working ports were placed in the axillary and back region. The pleura was incised, and the azygos vein was divided with ultrasonic coagulating shears. The tracheoesophageal fistula was dissected and sectioned after being closed with two 5-mm titanium clips. Anastomosis was done with 8 to 9 interrupted stitches using extracorporeal knot-tying techniques.
1.3. Esophageal videomanometry Videomanometry with topographic analysis was conducted to investigate esophageal motor function. The details of this procedure have been reported previously [7,8]. Briefly, a 16-channel side-hole assembly with 1-cm intervals between each side-hole was used with a low compliance Dentsleeve pump MKII (Dentsleeve Pty, SA, Australia). The locations of side-holes were determined to cover an area from the stomach to the upper esophageal body. The digital pressure signals were transferred to a personal computer concurrently with fluoroscopic images consecutively obtained. Recordings were made with several swallows of contrast medium (1 mL of iopamidol). Videomanometry provided luminal closure fluoroscopic images proceeding down the esophagus with clearance of the contrast medium and the coordinated motor pattern of the esophagus. Topographic plots were derived by aligning pressure data from the recording sites. Contour plots displayed 3dimensional data with concentric rings of higher amplitude indicating regional peaks in the contraction profile. Topographic plotting images showed an esophageal contraction sequence at swallow, which was formed by a chain of sequential pressure events. The chain was normally composed of 4 segments separated by 3 pressure troughs [7].
1.4. Twenty-four–hour esophageal pH monitoring Under fluoroscopic control, an antimony pH electrode (Medtronics) located at the tip of the pH probe was placed a vertebral body length above the line between the bilateral cranial peak points of the diaphragmatic dome to measure distal esophageal pH. pH values were recorded every 4 seconds for 24 hours and stored on a portable data logger (Diggitrapper Mark III, Medtronics, Minneapolis, MN). All data were then downloaded to a computer for analysis using dedicated software, EsopHogram Reflux Analysis (Medtronics). All, except case 7, in group A received videomanometry and 24-hour esophageal pH monitoring. Because case 7 in group A showed advanced cardiorespiratory distress requir-
H. Kawahara et al. ing tracheostomy, the operative indication of fundoplication was determined by clinical and fluoroscopic findings. Videomanometry was conducted at the age of 47 days (range, 28-116 days) and 39 days (range, 24-127 days) in groups A and B, respectively (P = .92). Twenty-four–hour esophageal pH monitoring was conducted at the age of 41 days (range, 31-112 days) and 39 days (range, 24-119 days) in groups A and B, respectively (P = .87). Case 7 in group A died of advanced cardiopulmonary failure at the age of 30 months after moving to another hospital at the age of 21 months. The remaining patients except case 7 in group B were followed up. Case 7 in group B moved away at the age of 6 months because of socioeconomic reasons. The follow-up period of the patients excluding the above 2 patients showed median values of 51 months (range, 24-75 months) and 51 months (range, 43-70 months) in groups A and B, respectively (P = .72). All data are expressed as medians and ranges. The MannWhitney U test and Fisher exact probability test were used for statistical analysis of data between groups A and B, with P b .05 considered as significant.
2. Results The individual data are detailed in Table 2. Topographic analysis showed an absence of the second segment in the chain of sequential pressure events, indicating a lack of esophageal contractions in the anastomotic region in all patients. Esophageal motor patterns shown by topographic analysis were classified into 2 groups according to the presence or absence of the third segment in the chain of sequential pressure events, which indicates contractions in the distal esophagus. The third segment was conspicuously absent in 1 and 3 patients in groups A and B, respectively (P = .26). In addition, the contraction patterns in the distal esophagus were classified into 2 types: peristaltic and synchronous. Peristaltic contractions were observed in 7 (78%) and 1 (14%) patients in groups A and B, respectively (P = .04). The median values of esophageal acid exposure measured by 24-hour esophageal pH monitoring were 5.5% (range, 0.7%-24.6%) and 3.7% (range, 0.3%-56.8%) in groups A and B, respectively (P = .71). The median values of the mean esophageal acid reflux time were 0.5 minutes (range, 0.1-1.4 minutes) and 0.5 minutes (range, 0.1-1.3 minutes) in groups A and B, respectively (P = .87). Fundoplication was done to control frequent pneumonia/hematemesis in 2 patients in the 2 groups (P = .60). Of the 4 patients undergoing fundoplication, 3 were associated with advanced neurologic impairment, and all 3 patients with videomanometry showed conspicuously absent distal esophageal contractions. The remaining 13 patients did not require fundoplication or aggressive medical treatment for GER. Prophylactic oral administration of famotidine was continued until the age of 3 months in 2 patients in group
Thoracoscopic esophageal atresia repair and GER Table 2
Results of each subject
Case
Videomanometry
Distal esophageal pH study
Distal esophageal contractions
% time pH b4.0
Average reflux time (min)
0.7 21.1
0.1 1.4
4.9 24.6 5.5 2.7 –
0.5 0.8 0.6 0.3 –
20.2 5.8 1.0
0.6 0.3 0.1
33.9 56.8 4.0 0.9 3.7 2.8 0.3
1.3 0.7 0.3 0.8 0.2 0.5 0.1
Group A (open repair) 1 Peristaltic 2 Absent 3 4 5 6 7
Peristaltic Peristaltic Peristaltic Synchronous Not examined
8 Peristaltic 9 Peristaltic 10 Peristaltic Group B (thoracoscopic repair) 1 Absent 2 Absent 3 Synchronous 4 Synchronous 5 Peristaltic 6 Synchronous 7 Absent
2285
Prophylactic H2 blocker
Fundoplication Age at operation
Operative indications
+
11 mo
Hematemesis, repeated pneumonia
+
7 mo
Respiratory distress, frequent emesis
+ +
23 mo 4 mo
Repeated pneumonia ALTE
3 mo 3 mo
3 mo 6 mo
ALTE indicates apparent life-threatening event.
A and 1 in group B. Case 7 in group B was given famotidine orally until the age of 6 months because an upper gastrointestinal study showed free reflux, and 3 sessions of esophageal dilatations were necessary to treat intractable esophageal anastomotic stricture, although she did not have frequent emesis or excessive distal esophageal acid exposure.
3. Discussion Recent advancements in techniques and instrumentation in pediatric endoscopic surgery have allowed significantly more complex and delicate procedures to be performed. Lobe et al [2] did the first minimally invasive thoracoscopic repair of EA almost 60 years after the first successful primary repair. Although aesthetically superior to open repair, this minimally invasive technique requires advanced endosurgical and anesthetic expertise [5]. A question was raised as to whether this technique is better than the traditional open approach [4]. This technique has been reported to allow very precise suturing because of the superior visualization, which is thought to result in less motility disturbance [5]. However, whether thoracoscopic dissection has a less detrimental effect on disturbed motility remains to be proven [5]. Therefore, this retrospective analysis was conducted to clarify the advantage of this novel technique. The selection of 2 operative procedures was not standardized in this retrospective study, which was deter-
mined with multiple factors, mainly the patient's birth weight, associated anomalies, and general condition. There were no significant differences in terms of patient characteristics between the 2 groups, except birth weight. The peristaltic pattern of esophageal contractions was observed more frequently in patients with open repair, whereas pH data and the incidence of fundoplication were not different between the 2 groups. Esophageal anastomotic stricture requiring balloon dilatation was observed in only 2 patients with thoracoscopic repair. These findings may not support the thought that the minimally invasive approach contributes to a reduction in the disturbance of esophageal motor function, which is definitely of long-term importance to patients with EA. Of 4 patients with conspicuously absent distal esophageal contractions, 3 patients underwent fundoplication because of intractable GER-related symptoms. The remaining patient showed esophageal anastomotic stricture, although excessive esophageal acid exposure was not demonstrated with 24-hour pH monitoring. As reported previously [8], this study also showed that the virtual absence of contractions in the distal esophagus, which indicates lack of clearing capacity of reflux material, had a significant impact on the development of symptomatic GER. Our finding that thoracoscopic repair did not influence the occurrence of subsequent symptomatic GER is comparable to recent multiinstitutional analysis showing a 24% incidence of fundoplication in patients with thoracoscopic repair [4]. Therefore, disorders of esophageal motor function may result more from
2286 an inherent abnormal innervation of the esophagus than from intraoperative denervation [9]. This retrospective and limited-number study failed to reveal the advantage of thoracoscopic repair in terms of postoperative esophageal motor function. Because this technique is an extremely attractive alternative to the traditional open approach, prospective and randomized studies are necessary to clarify the long-term benefits for patients with EA undergoing this technique.
References [1] Goyal A, Jones MO, Couriel JM, et al. Oesophageal atresia and tracheooesophageal fistula. Arch Dis Child 2006;91:F381-4. [2] Lobe TE, Rothenberg SS, Waldschmidt J, et al. Thoracoscopic repair of esophageal atresia in an infant: a surgical first. Pediatr Endosurg Innovative Tech 1999;3:141-8.
H. Kawahara et al. [3] Rothenberg SS. Thoracoscopic repair of tracheoesophageal fistula in newborns. J Pediatr Surg 2002;37:869-72. [4] Holcomb III GW, Rothenberg SS, Bax KM, et al. Thoracoscopic repair of esophageal atresia and tracheoesophageal fistula: a multi-institutional analysis. Ann Surg 2005;242:422-30. [5] van der Zee DC, Bax KN. Thoracoscopic treatment of esophageal atresia with distal fistula and of tracheomalacia. Semin Pediatr Surg 2007;16:224-30. [6] Okuyama H, Kubota A, Kawahara H, et al. Thoracoscopic repair of a tracheoesophageal fistula in a newborn infant. J Jpn Soc Pediatr Surg 2005;41:23-7. [7] Kawahara H, Kubota A, Okuyama H, et al. The usefulness of videomanometry for studying pediatric esophageal motor disease. J Pediatr Surg 2004;39:1754-7. [8] Kawahara H, Kubota A, Hasegawa T, et al. Lack of distal esophageal contractions is a key determinant of gastroesophageal reflux disease after repair of esophageal atresia. J Pediatr Surg 2007; 42:2017-21. [9] Nakazato Y, Landing BH, Wells TR. Abnormal Auerbach plexus in the esophagus and stomach of patients with esophageal atresia and tracheoesophageal fistula. J Pediatr Surg 1986;21:831-7.
www.elsevier.com/locate/jpedsurg
Influence of thoracoscopic esophageal atresia repair on esophageal motor function and gastroesophageal reflux☆ Hisayoshi Kawaharaa,⁎, Hiroomi Okuyamaa , Yasuyuki Mitania , Motonari Nomuraa , Keisuke Nosea , Akihiro Yonedaa , Toshimichi Hasegawaa , Akio Kubotaa , Masahiro Fukuzawab a
Department of Pediatric Surgery, Osaka Medical Center and Research Institute for Maternal and Child Health, Osaka 594-1101, Japan b Department of Pediatric Surgery, Osaka University Graduate School of Medicine, Osaka, 565-0871, Japan Received 20 July 2009; accepted 31 July 2009
Key words: Esophageal atresia; Gastroesophageal reflux; Thoracoscopic repair; Videomanometry; 24-Hour esophageal pH monitoring
Abstract Background/Purpose: Thoracoscopic repair has recently been attempted in newborns with esophageal atresia (EA), but it remains unclear whether thoracoscopic dissection reduces pathological gastroesophageal reflux. We investigated the influence of a thoracoscopic approach on esophageal motor function in patients with EA. Methods: Clinical and gastrointestinal data of 10 patients with EA with open repair (group A) and 7 with thoracoscopic repair (group B) were analyzed retrospectively. Videomanometry was conducted to investigate esophageal motor patterns. Esophageal acid exposure was evaluated with 24-hour esophageal pH monitoring. Data are expressed as medians and ranges. Results: Contractions in the distal esophagus were conspicuously absent in 1 and 3 patients in groups A and B, respectively (P = .26). There were no significant differences in esophageal acid exposure (5.5% [0.7%-24.6%] vs 3.7% [0.3%-56.8%]; P = .71) or mean esophageal acid reflux time (0.5 minutes [0.11.4 minutes] vs 0.5 minutes [0.1-1.3 minutes]; P = .87) between the 2 groups. Fundoplication was conducted in 2 patients in each group (P = .60), all of whom had conspicuously absent distal esophageal contractions. Those contractions were preserved in the remaining patients with the exception of 1 group B patient. Conclusion: There are unlikely to be benefits from thoracoscopic repair of EA in terms of postoperative esophageal motor function. © 2009 Elsevier Inc. All rights reserved.
Presented at the 42nd Annual Meeting of the Pacific Association of Pediatric Surgeons, Hong Kong, China, May 10-14, 2009. ☆ Hisayoshi Kawahara, MD, was supported by the Sukoyaka Grant for Maternal and Child Health. ⁎ Corresponding author. Tel.: +81 725 56 1220; fax: +81 725 56 5682. E-mail address: [email protected] (H. Kawahara). 0022-3468/$ – see front matter © 2009 Elsevier Inc. All rights reserved. doi:10.1016/j.jpedsurg.2009.07.049
The emphasis of esophageal atresia (EA) in the modern era has changed to reducing morbidity and improving the quality of life after correction of EA [1]. Since the first introduction by Rothenberg et al, minimal invasive thoracoscopic repair of EA has been conducted in advanced pediatric endosurgical centers [2-5]. It is believed to be a superior cosmetic method with fewer sequelae, including
Thoracoscopic esophageal atresia repair and GER thoracic deformities and scoliosis. However, it remains to be proven whether thoracoscopic dissection has a less detrimental effect on esophageal motor function, which possibly contributes to preventing pathological gastroesophageal reflux (GER) occurring after repair of EA [5]. We aimed to investigate the influence of a thoracoscopic approach on esophageal motor function in patients with EA.
1. Materials and methods A retrospective review of 17 consecutive patients with Gross type C EA undergoing surgical correction with primary esophageal anastomosis in the period between December 2002 and April 2007 was performed. Data collected from the medical records included birth weight, gestational age, prenatal diagnosis, associated anomalies, operative procedure, postoperative leak, and stricture, as well as esophageal dilatation for anastomotic stricture if conducted. In addition, data obtained with videomanometry and 24-hour esophageal pH study were analyzed. These examinations were conducted as routine follow-up studies or as required by a patient's clinical condition. Ethical approval of these exminations was obtained from the
Table 1 Case
departmental committee, and informed consent was obtained from the patients' guardians. Administration of H2-receptor antagonists and fundoplication were also recorded if used.
1.1. Patients with EA The characteristics of each patient are detailed in Table 1. Of the 17 patients, 10 underwent open repair of EA (group A) and 7 thoracoscopic repair (group B). There was a significant difference in birth weight between groups A and B (2450 g [range, 1464-2908 g] vs 2814 g [range, 2460-3710 g]; P = .02) but no significant difference in gestational age (38 weeks [range, 33-41 weeks] vs 40 weeks [range, 37-41 weeks]; P = .15). The incidences of prenatal diagnosis (P = .33), association with other anomalies (P N .99), and gastrostomy (P = .30) were not significantly different between the 2 groups. The age at primary repair of EA was 2 days (range, 012 days) and 1 day (range, 1-3 days) in groups A and B, respectively (P = .37). The incidence of a minor leak was not significantly different between the 2 groups (P N .99), and only case 3 in group B showed fever and C-reactive protein (CRP) elevation. Two patients in group B, but none in group A, required esophageal balloon dilatation (P = .15).
Characteristics of each subject Birth weight (g)
Gestational age (wk)
Group A (open repair) 1 2908 41 2 2042 37 3 2626 37 4 1464 33 5 2580 38 6 2411 41 7 1610 34
8 9
2283
2170 2900
36 40
10 2488 39 Group B (thoracoscopic repair) 1 2814 41
Prenatal diagnosis
− + − − − − +
− +
Associated anomalies
CHARGE association, ECD Laryngeal atresia
Dudenal atresia, VSD Polydactyly, tracheomalacia VACTER association, tetralogy of fallot, duodenal atresia, severe scoliosis Agenesis of right kidney DORV, duodenal atresia, rectourethral fistula
Gastrostomy
2
2664
37
+
3 4 5 6 7
2460 3710 2604 2988 3046
40 41 41 38 38
− − + + +
Treacher Collins syndrome, micrognathia CHARGE association, VSD, cerebellar hypoplasia, hydronephrosis
Postoperative upper gastrointestinal Leak
Stricture
+
8 3 1 3 1 1 12
− Minor Minor − − − −
− − − − − − −
+
1 3
Minor −
− −
0
−
−
1
Minor
−
3
−
−
1 1 1 1 2
Minor − − − −
+ − − − +
+ + +
+ +
Age at primary repair (d)
+
Duodenal atresia, VSD Hydronephrosis Rectovestibular fistula
ECD, endocardial cushion defect; VSD, ventricular septal defect; DORV, double outlet right ventricle.
2284
1.2. Thoracoscopic repair of EA All thoracoscopic repair was conducted by one of the authors (HO) [6]. The patient was positioned prone with the right side slightly elevated. A 3-cm camera port was inserted into the sixth intercostal space. The pleural space was insufflated to a pressure of 3 to 4 mm Hg. Additional 3 working ports were placed in the axillary and back region. The pleura was incised, and the azygos vein was divided with ultrasonic coagulating shears. The tracheoesophageal fistula was dissected and sectioned after being closed with two 5-mm titanium clips. Anastomosis was done with 8 to 9 interrupted stitches using extracorporeal knot-tying techniques.
1.3. Esophageal videomanometry Videomanometry with topographic analysis was conducted to investigate esophageal motor function. The details of this procedure have been reported previously [7,8]. Briefly, a 16-channel side-hole assembly with 1-cm intervals between each side-hole was used with a low compliance Dentsleeve pump MKII (Dentsleeve Pty, SA, Australia). The locations of side-holes were determined to cover an area from the stomach to the upper esophageal body. The digital pressure signals were transferred to a personal computer concurrently with fluoroscopic images consecutively obtained. Recordings were made with several swallows of contrast medium (1 mL of iopamidol). Videomanometry provided luminal closure fluoroscopic images proceeding down the esophagus with clearance of the contrast medium and the coordinated motor pattern of the esophagus. Topographic plots were derived by aligning pressure data from the recording sites. Contour plots displayed 3dimensional data with concentric rings of higher amplitude indicating regional peaks in the contraction profile. Topographic plotting images showed an esophageal contraction sequence at swallow, which was formed by a chain of sequential pressure events. The chain was normally composed of 4 segments separated by 3 pressure troughs [7].
1.4. Twenty-four–hour esophageal pH monitoring Under fluoroscopic control, an antimony pH electrode (Medtronics) located at the tip of the pH probe was placed a vertebral body length above the line between the bilateral cranial peak points of the diaphragmatic dome to measure distal esophageal pH. pH values were recorded every 4 seconds for 24 hours and stored on a portable data logger (Diggitrapper Mark III, Medtronics, Minneapolis, MN). All data were then downloaded to a computer for analysis using dedicated software, EsopHogram Reflux Analysis (Medtronics). All, except case 7, in group A received videomanometry and 24-hour esophageal pH monitoring. Because case 7 in group A showed advanced cardiorespiratory distress requir-
H. Kawahara et al. ing tracheostomy, the operative indication of fundoplication was determined by clinical and fluoroscopic findings. Videomanometry was conducted at the age of 47 days (range, 28-116 days) and 39 days (range, 24-127 days) in groups A and B, respectively (P = .92). Twenty-four–hour esophageal pH monitoring was conducted at the age of 41 days (range, 31-112 days) and 39 days (range, 24-119 days) in groups A and B, respectively (P = .87). Case 7 in group A died of advanced cardiopulmonary failure at the age of 30 months after moving to another hospital at the age of 21 months. The remaining patients except case 7 in group B were followed up. Case 7 in group B moved away at the age of 6 months because of socioeconomic reasons. The follow-up period of the patients excluding the above 2 patients showed median values of 51 months (range, 24-75 months) and 51 months (range, 43-70 months) in groups A and B, respectively (P = .72). All data are expressed as medians and ranges. The MannWhitney U test and Fisher exact probability test were used for statistical analysis of data between groups A and B, with P b .05 considered as significant.
2. Results The individual data are detailed in Table 2. Topographic analysis showed an absence of the second segment in the chain of sequential pressure events, indicating a lack of esophageal contractions in the anastomotic region in all patients. Esophageal motor patterns shown by topographic analysis were classified into 2 groups according to the presence or absence of the third segment in the chain of sequential pressure events, which indicates contractions in the distal esophagus. The third segment was conspicuously absent in 1 and 3 patients in groups A and B, respectively (P = .26). In addition, the contraction patterns in the distal esophagus were classified into 2 types: peristaltic and synchronous. Peristaltic contractions were observed in 7 (78%) and 1 (14%) patients in groups A and B, respectively (P = .04). The median values of esophageal acid exposure measured by 24-hour esophageal pH monitoring were 5.5% (range, 0.7%-24.6%) and 3.7% (range, 0.3%-56.8%) in groups A and B, respectively (P = .71). The median values of the mean esophageal acid reflux time were 0.5 minutes (range, 0.1-1.4 minutes) and 0.5 minutes (range, 0.1-1.3 minutes) in groups A and B, respectively (P = .87). Fundoplication was done to control frequent pneumonia/hematemesis in 2 patients in the 2 groups (P = .60). Of the 4 patients undergoing fundoplication, 3 were associated with advanced neurologic impairment, and all 3 patients with videomanometry showed conspicuously absent distal esophageal contractions. The remaining 13 patients did not require fundoplication or aggressive medical treatment for GER. Prophylactic oral administration of famotidine was continued until the age of 3 months in 2 patients in group
Thoracoscopic esophageal atresia repair and GER Table 2
Results of each subject
Case
Videomanometry
Distal esophageal pH study
Distal esophageal contractions
% time pH b4.0
Average reflux time (min)
0.7 21.1
0.1 1.4
4.9 24.6 5.5 2.7 –
0.5 0.8 0.6 0.3 –
20.2 5.8 1.0
0.6 0.3 0.1
33.9 56.8 4.0 0.9 3.7 2.8 0.3
1.3 0.7 0.3 0.8 0.2 0.5 0.1
Group A (open repair) 1 Peristaltic 2 Absent 3 4 5 6 7
Peristaltic Peristaltic Peristaltic Synchronous Not examined
8 Peristaltic 9 Peristaltic 10 Peristaltic Group B (thoracoscopic repair) 1 Absent 2 Absent 3 Synchronous 4 Synchronous 5 Peristaltic 6 Synchronous 7 Absent
2285
Prophylactic H2 blocker
Fundoplication Age at operation
Operative indications
+
11 mo
Hematemesis, repeated pneumonia
+
7 mo
Respiratory distress, frequent emesis
+ +
23 mo 4 mo
Repeated pneumonia ALTE
3 mo 3 mo
3 mo 6 mo
ALTE indicates apparent life-threatening event.
A and 1 in group B. Case 7 in group B was given famotidine orally until the age of 6 months because an upper gastrointestinal study showed free reflux, and 3 sessions of esophageal dilatations were necessary to treat intractable esophageal anastomotic stricture, although she did not have frequent emesis or excessive distal esophageal acid exposure.
3. Discussion Recent advancements in techniques and instrumentation in pediatric endoscopic surgery have allowed significantly more complex and delicate procedures to be performed. Lobe et al [2] did the first minimally invasive thoracoscopic repair of EA almost 60 years after the first successful primary repair. Although aesthetically superior to open repair, this minimally invasive technique requires advanced endosurgical and anesthetic expertise [5]. A question was raised as to whether this technique is better than the traditional open approach [4]. This technique has been reported to allow very precise suturing because of the superior visualization, which is thought to result in less motility disturbance [5]. However, whether thoracoscopic dissection has a less detrimental effect on disturbed motility remains to be proven [5]. Therefore, this retrospective analysis was conducted to clarify the advantage of this novel technique. The selection of 2 operative procedures was not standardized in this retrospective study, which was deter-
mined with multiple factors, mainly the patient's birth weight, associated anomalies, and general condition. There were no significant differences in terms of patient characteristics between the 2 groups, except birth weight. The peristaltic pattern of esophageal contractions was observed more frequently in patients with open repair, whereas pH data and the incidence of fundoplication were not different between the 2 groups. Esophageal anastomotic stricture requiring balloon dilatation was observed in only 2 patients with thoracoscopic repair. These findings may not support the thought that the minimally invasive approach contributes to a reduction in the disturbance of esophageal motor function, which is definitely of long-term importance to patients with EA. Of 4 patients with conspicuously absent distal esophageal contractions, 3 patients underwent fundoplication because of intractable GER-related symptoms. The remaining patient showed esophageal anastomotic stricture, although excessive esophageal acid exposure was not demonstrated with 24-hour pH monitoring. As reported previously [8], this study also showed that the virtual absence of contractions in the distal esophagus, which indicates lack of clearing capacity of reflux material, had a significant impact on the development of symptomatic GER. Our finding that thoracoscopic repair did not influence the occurrence of subsequent symptomatic GER is comparable to recent multiinstitutional analysis showing a 24% incidence of fundoplication in patients with thoracoscopic repair [4]. Therefore, disorders of esophageal motor function may result more from
2286 an inherent abnormal innervation of the esophagus than from intraoperative denervation [9]. This retrospective and limited-number study failed to reveal the advantage of thoracoscopic repair in terms of postoperative esophageal motor function. Because this technique is an extremely attractive alternative to the traditional open approach, prospective and randomized studies are necessary to clarify the long-term benefits for patients with EA undergoing this technique.
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H. Kawahara et al. [3] Rothenberg SS. Thoracoscopic repair of tracheoesophageal fistula in newborns. J Pediatr Surg 2002;37:869-72. [4] Holcomb III GW, Rothenberg SS, Bax KM, et al. Thoracoscopic repair of esophageal atresia and tracheoesophageal fistula: a multi-institutional analysis. Ann Surg 2005;242:422-30. [5] van der Zee DC, Bax KN. Thoracoscopic treatment of esophageal atresia with distal fistula and of tracheomalacia. Semin Pediatr Surg 2007;16:224-30. [6] Okuyama H, Kubota A, Kawahara H, et al. Thoracoscopic repair of a tracheoesophageal fistula in a newborn infant. J Jpn Soc Pediatr Surg 2005;41:23-7. [7] Kawahara H, Kubota A, Okuyama H, et al. The usefulness of videomanometry for studying pediatric esophageal motor disease. J Pediatr Surg 2004;39:1754-7. [8] Kawahara H, Kubota A, Hasegawa T, et al. Lack of distal esophageal contractions is a key determinant of gastroesophageal reflux disease after repair of esophageal atresia. J Pediatr Surg 2007; 42:2017-21. [9] Nakazato Y, Landing BH, Wells TR. Abnormal Auerbach plexus in the esophagus and stomach of patients with esophageal atresia and tracheoesophageal fistula. J Pediatr Surg 1986;21:831-7.