Journal of Pediatric Surgery (2005) 40, 781 – 784
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Multistaged esophageal elongation technique for long gap esophageal atresia: experience with 7 cases at a single institution Shigeru Takamizawa*, Eiji Nishijima, Chikara Tsugawa, Toshihiro Muraji, Shiiki Satoh, Yukihiro Tatekawa, Ken Kimura Department of Surgery, Kobe Children’s Hospital, Kobe 654-0081, Japan Index words: Multistaged esophageal elongation; Esophageal atresia; Long gap; Native esophagus
Abstract Background/Purpose: Esophageal reconstruction for long gap esophageal atresia (LGEA) is still controversial. We successfully managed 7 cases of patients with LGEA by doing staged elongation of the native esophagus and subsequent end-to-end anastomosis. The technique and efficacy of this procedure are evaluated. Methods: During the last 10 years, 7 patients with LGEA (Gross type A, 5; B, 1; C, 1) underwent multiple extrathoracic esophageal elongations (ETEEs) of the upper esophagus and subsequent esophagoesophagostomy. Medical records were reviewed in regard to the number of ETEE before definitive esophageal reconstruction, interval between each ETEE, operation time, time before initiation of sham feeding, duration of hospital stay, and complications. Results: The definitive esophageal reconstruction was successfully achieved without major complications in all patients after 2 to 4 stages of ETEE. The interval between each ETEE was 72 days on average. The average operation time was 98 minutes. The elongation was 1 to 3.5 cm during each session. Oral sham feeding was recommenced 4.1 days after each ETEE, and the hospital stay was 9.6 days on average. Gastroesophageal reflux occurred in all patients, requiring antireflux surgery. Conclusions: We conclude from our experience (a) that effective esophageal lengthening with preservation of the native esophagus was achieved with multiple ETEE in LGEA and (b) that this procedure allows oral sham feeding at home until esophageal reconstruction. D 2005 Elsevier Inc. All rights reserved.
Surgical procedure for patients with long gap esophageal atresia (LGEA) is still controversial. Several techniques have been introduced. However, the ideal procedure to bridge the gap between the proximal and the distal esophagus is as yet Presented at the 36th Annual Meeting of the Canadian Association of Pediatric Surgeons, Winnipeg, Manitoba, Canada, September 30October 3, 2004. T Corresponding author. Tel.: +81 78 732 6961; fax: +81 78 735 0910. E-mail address:
[email protected] (S. Takamizawa). 0022-3468/$ – see front matter D 2005 Elsevier Inc. All rights reserved. doi:10.1016/j.jpedsurg.2005.01.041
undetermined. We used a multistaged extrathoracic esophageal elongation (ETEE) technique to treat our patients with LGEA and evaluated the efficacy of this technique.
1. Materials and methods Between March 1994 and January 2004, we treated 7 patients with LGEA at our institution. Medical charts were reviewed retrospectively, and patient characteristics are
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Table 1
Patient characteristics
Case No. Type
1 2 3 4 5a 6a 7
Gross Gross Gross Gross Gross Gross Gross a
Gap measurement (cm) First elongation
A A A A C A B
3.5 4 3 3 6.5 5 3.8
Second elongation
Third elongation
Fourth elongation
Esophagoesophagostomy
Age Weight Age Weight Age Weight Age Weight Age (mo) (kg) (mo) (kg) (mo) (kg) (mo) (kg) (mo)
Weight (kg)
5 10 1 2 5 20 4
5.4 8.4 7.3 5.3 13 7.7 8.4
4.8 7.8 2.8 3.4 8.7 6.6 4.8
7 12 3 3 9 22 7
5.2 8 3.8 4.1 10.5 7.2 5.8
5 11 25 10
4.7 10.9 7.9 7.3
14
11
8 16 12 7 17 27 13
Initial cutaneous esophagostomy was created in another hospital.
summarized in Table 1. Although 4 of 7 patients were previously reported by Nishijima et al [1] and Kimura et al [2], we include them in this report because they provide important information of a long-term follow-up. Five patients had pure esophageal atresia (EA) without tracheoesophageal fistula (Gross type A), one had EA with proximal tracheoesophageal fistula (Gross type B), and another had EA with distal tracheoesophageal fistula (Gross type C) with failed primary anastomosis who underwent diverting cervical esophagostomy in another hospital. We reviewed these patients in regard to the number of ETEE, interval between each ETEE, operation time of ETEE, time to initiate sham feeding after each ETEE, duration of hospital stay, and operative complications.
In the definitive operation, the esophagus is dissected to the cricoid cartilage in the same fashion as each elongation. The route where the upper esophagus passes is created between the trachea and the cervical vertebra (anterolateral to the cervical vertebra) to the right thoracic cavity. The cervical and anterior chest incisions are closed temporarily, and a right thoracotomy is performed for the esophageal anastomosis. Excision of the scarred distal end of the upper esophagus is mandatory to minimize postoperative anastomotic stenosis. The distal esophageal pouch is then mobilized. If an end-to-end esophageal anastomosis is not possible, a laparotomy is performed and lengthening of the lower esophagus is accomplished by Collis procedure with Nissen fundoplication (Collis-Nissen fundoplication) [4].
1.1. Cutaneous esophagostomy, subsequent ETEE, and definitive esophageal reconstruction The operative procedure consisting of initial cutaneous esophagostomy, ETEE, and definitive esophageal anastomosis was introduced by Kimura et al [2] and Kimura and Soper [3]. At the initial cutaneous esophagostomy, a transverse skin incision is made above the right clavicle. The sternal head of the sternocleidomastoid muscle is retracted laterally or divided, and the proximal esophageal pouch is mobilized. Another transverse skin incision is made on the right anterior chest, creating a subcutaneous tunnel through which the proximal esophagus is passed and exteriorized as an esophagostomy. After a few months, the first ETEE is performed (Fig. 1). The esophagostomy is mobilized and dissected up to the cricoid cartilage level. Variable elongation of the esophagus is achieved after the completion of the dissection. This is then again exteriorized as an esophagostomy a few centimeters below the previous esophagostomy site. Each ETEE is scheduled every 2 to 3 months until the esophageal length is deemed sufficient to allow restoration of esophageal continuity. Oral sham feeding is commenced 2 to 3 days after each ETEE. The patients are discharged on gastrostomy button and oral sham feeding until the time of the next ETEE or definitive operation.
Fig. 1 ETEE: the esophagostomy is mobilized and dissected up to the cricoid cartilage level, with the opening placed on a previous superior clavicle surgical scar. Variable elongation of the esophagus is achieved after completion of the dissection. This is then again exteriorized as an esophagostomy at a level a few centimeters below the previous esophagostomy site.
Multistaged esophageal elongation technique for long- gap esophageal atresia
2. Results Two to 4 stages of ETEE were required before definitive intrathoracic esophageal reconstruction (Table 1). The mean interval between each ETEE was 72 days (range, 31-201 days). The average operation time of ETEE was 98 minutes (range, 60-135 minutes). In each ETEE, an increase in the upper esophageal length of 1 to 3.5 cm was achieved. Oral sham feeding was started at the 4.1 day after ETEE (range, 1-14 days). The average hospital stay after ETEE was 9.6 days (range, 4-22 days except patient 1 who stayed in the hospital until the definitive esophageal reconstruction was performed). Patient 1 developed wound infection, which was treated conservatively, resulting in a stenosis of the esophagostomy. Stenosis was corrected during the next ETEE. Wound dehiscence, which was managed conservatively, was observed in case 7. Definitive esophagoesophagostomy was successfully performed in all 7 patients at 7 to 27 months of age. Anastomotic leakage was observed in 2 patients; both were managed conservatively. All patients had anastomotic stricture which required balloon dilatation (range, 1-13 times). Esophageal perforation during balloon dilatation occurred twice in case 4. On the first occurrence, only drainage was required. The second occurrence required an operative perforation repair. This patient underwent resection of the stenotic part with reanastomosis of the esophagus 3 months after the second perforation. All 7 patients required a fundoplication. Five of 7 patients (Gross type A: 4 cases, Gross type B: 1 case) underwent a partial Nissen fundoplication for gastroesophageal reflux after the definitive esophageal reconstruction. The other 2 patients (Gross type A: 1 case, Gross type C: 1 case) in whom an esophagoesophagostomy had failed, underwent Collis-Nissen fundoplication at the time of definitive operation because of shortening of the lower esophagus. All patients in this study are doing well without eating difficulty after a mean follow-up period of 4.5 years (range, 0.3-9.8 years) after the last operation.
3. Discussion Historically, several procedures for treating LGEA were reported and these are categorized as follows: (1) primary anastomosis [5], (2) delayed primary anastomosis after preoperative bouginage [6,7], (3) intraoperative myotomy of upper esophageal pouch [8,9], (4) Foker’s 2-staged primary repair [10], (5) multistaged ETEE procedure [2,3], or (6) bridging the esophagus using stomach, jejunum, or colon [11-19]. However, the native esophagus is still considered superior to the esophageal replacement, and every effort should be made to restore continuity between the upper and lower esophageal pouches. By a simple bouginage however, patients must remain in the hospital to allow appropriate suctioning of accumulated saliva in the upper esophageal pouch. Oral sham feeding has inevitably
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to be abandoned. Early initiation and continuation of oral feeding is important to avoid the subsequent feeding difficulty and oral aversion in the treatment of EA. The multistaged ETEE technique first reported by Kimura et al [2] and Kimura and Soper [3] resolved these 3 important problems in treating patients with LGEA. This technique uses the patients’ native esophagus, which is thought to be the best organ to reconstruct the esophagus, allows early initiation of oral sham feedings, and shortens hospital stay. We used this technique to manage pure EA and applied it to a patient who failed in an attempt esophageal reconstruction for EA with distal tracheoesophageal fistula. In this patient, the distal esophagus had been resected. The patient underwent an ETEE 4 times, which allowed a successful definitive esophageal reconstruction. Two and a half years after the esophageal reconstruction, the patient is able to eat any table food without difficulty. In 4 patients who were previously reported, 1 patient developed an esophageal perforation twice while balloon dilatation was being performed [1,2]. The first time, only drainage was required, however, on the second time, a surgical closure and consequent resection and reanastomosis were required (case 4). Three remaining patients are free of feeding difficulties except 1 patient (case 1) who underwent balloon dilatation after the previous report. Adhesion of the esophagus to the soft tissue and ischemic change at the stretched distal end of the elongated proximal esophagus are the concerns for performing ETEE procedures. Dessanti et al [20] used Gore-Tex membrane (WL Gore and Associates, Flagstaff, Ariz) to minimize surgical adhesions in ETEE. Although we have not used any prosthesis, we never encountered a difficulty in dissecting the esophagus from the surrounding tissue. Blood supply to the distal end of the upper esophagus is presumably maintained by intramural-developed collaterals from the inferior thyroid artery and by collaterals from the vessels nourishing the subcutaneous tissue. Stenosis in the esophagostomy or cicatrization at the distal part of the upper esophagus because of local ischemia or tissue injury by multiple procedures could be happening. Necrosis of the elongated esophagus is a possible devastating complication, but no report has been encountered. Another concern is an anastomotic stricture. In our patients, an esophageal balloon dilatation was required 7 times on average for each patient. High tension, insufficient discarding of the scarred part of the esophagus, and gastroesophageal reflux are the contributing factors to anastomotic stricture. To minimize anastomotic tension, sufficient elongation is mandatory before the definitive operation. Thorough excision of cicatrized tissue is important for the successful anastomosis. We believe that ETEE technique is an effective procedure for LGEA that enables us to use the native esophagus. During the intervals of ETEE, patients can be cared for at home. Oral sham feeding preserves patients’ swallowing ability and avoids future eating difficulty and food aversion.
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