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Accepted Manuscript Thoracoscopic and laparoscopic esophagoplasty for congenital esophageal stenosis Naoto Urushihara, Hiroshi Nouso, Masaya Yamoto, Koji Fukumoto, Go Miyano, Hiromu Miyake, Keiichi Morita, Masakatsu Kaneshiro PII: S2213-5766(13)00140-1 DOI: 10.1016/j.epsc.2013.11.004 Reference: EPSC 139 To appear in: Journal of Pediatric Surgery Case Reports Received Date: 29 October 2013 Revised Date: 3 November 2013 Accepted Date: 5 November 2013 Please cite this article as: Urushihara N, Nouso H, Yamoto M, Fukumoto K, Miyano G, Miyake H, Morita K, Kaneshiro M, Thoracoscopic and laparoscopic esophagoplasty for congenital esophageal stenosis, Journal of Pediatric Surgery Case Reports (2013), doi: 10.1016/j.epsc.2013.11.004. This is a PDF file of an unedited manuscript that has been accepted for publication. As a service to our customers we are providing this early version of the manuscript. The manuscript will undergo copyediting, typesetting, and review of the resulting proof before it is published in its final form. Please note that during the production process errors may be discovered which could affect the content, and all legal disclaimers that apply to the journal pertain.

Thoracoscopic and laparoscopic esophagoplasty for congenital esophageal stenosis

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Accepted Manuscript

Thoracoscopic and laparoscopic esophagoplasty for congenital esophageal stenosis

Naoto Urushihara, Hiroshi Nouso, Masaya Yamoto, Koji Fukumoto, Go Miyano,Hiromu Miyake, Keiichi Morita, Masakatsu Kaneshiro

PII: S2213-5766(13)00140-1

DOI: 10.1016/j.epsc.2013.11.004

Reference: EPSC 139

To appear in: Journal of Pediatric Surgery Case Reports

Received Date: 29 October 2013

Revised Date: 3 November 2013

Accepted Date: 5 November 2013

Please cite this article as: Urushihara N, Nouso H, Yamoto M, Fukumoto K, Miyano G, Miyake H, MoritaK, Kaneshiro M, Thoracoscopic and laparoscopic esophagoplasty for congenital esophageal stenosis,Journal of Pediatric Surgery Case Reports (2013), doi: 10.1016/j.epsc.2013.11.004.

This is a PDF file of an unedited manuscript that has been accepted for publication. As a service toour customers we are providing this early version of the manuscript. The manuscript will undergocopyediting, typesetting, and review of the resulting proof before it is published in its final form. Pleasenote that during the production process errors may be discovered which could affect the content, and alllegal disclaimers that apply to the journal pertain.

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Revised manuscript:

Thoracoscopic and laparoscopic esophagoplasty for congenital esophageal stenosis

Naoto Urushihara, Hiroshi Nouso, Masaya Yamoto, Koji Fukumoto, Go Miyano, Hiromu Miyake,

Keiichi Morita, Masakatsu Kaneshiro

Department of Pediatric Surgery, Shizuoka Children’s Hospital, 860 Urushiyama, Aoi-ku, Shizuoka

420-8660, Japan

Address correspondence to:

Naoto Urushihara, MD, PhD

Department of Pediatric Surgery

Shizuoka Children’s Hospital

860 Urushiyama, Aoi-ku, Shizuoka 420-8660, Japan.

Phone: +81-54-247-6251 Fax: + 81-54-248-4763

E-mail: [email protected]

[email protected]

Short Running Head: Endoscopic esophagoplasty for congenital esophageal stenosis

Key words: Congenital esophageal stenosis; Laparoscopy; Thoracoscopy

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Abstract

A congenital esophageal stenosis (CES) is a rare anomaly, and therapeutic strategies are still

controversial. Two children with CES located in the lower esophagus, who were treated by

endoscopic esophagoplasty that consisted of partial esophageal resection and transverse anastomosis,

are reported. A 23-month-old boy with CES at the level of T9 underwent esophagoplasty

thoracoscopically. A 13-month-old boy with CES at the level of T10 underwent esophagoplasty

followed by anterior partial fundoplication laparoscopically. Both patients are asymptomatic and eat

normally 5 years and 6 months after surgery, respectively. Thoracoscopic or laparoscopic partial

resection of the esophageal wall and transverse anastomosis are considered effective, less invasive,

and safe in the treatment of CES.

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Congenital esophageal stenosis (CES) is a rare anomaly, with an incidence of about one per

25,000 to 50,000 live births [1]. CES is divided into 3 types: tracheobronchial remnants (TBR),

fibromuscular thickening (FM), and membranous webbing (MW). The therapeutic strategies,

including endoscopic balloon dilatation and resection of the stenotic site, are still controversial

[2][3][4][5][6][7][8]. Conventionally, esophageal resection has been performed via either

laparotomy or thoracotomy depending on the stenotic site. Recently, thoracoscopic or laparoscopic

surgeries have been reported for the treatment of CES [9][10][11][12]. The cases of two children

with CES located in the lower esophagus, who were treated by endoscopic esophagoplasty that

consisted of partial esophageal resection and transverse anastomosis, are described. A thoracoscopic

approach was used in one, and a laparoscopic approach in the other.

1. Case reports

A 23-month-old boy and a 13-month-old boy were admitted to our hospital for vomiting due to

foreign body impaction in the lower esophagus. The esophagogram and endoscopy showed a lower

esophageal stricture at the level of T9 and T10, respectively (Fig. 1). The proximal esophagus was

moderately dilated. Endoscopic ultrasonography (EUS) was performed to distinguish between TBR

and FM, and it did not show aberrant cartilage in both cases. The patients initially underwent balloon

dilatations 3 and 4 times, respectively, with no significant effect. Partial resections of the esophageal

wall were performed by less invasive surgery in these two children with CES. Histologic studies

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revealed FM in one and TBR in the other.

1.1 Case 1: 23-month-old boy with CES at the level of T9 due to FM

Partial esophageal resection and transverse anastomosis by a thoracoscopic approach were

performed for stenosis at the level of T9 (Fig. 1A). One-lung ventilation by right mainstem

intubation was carried out for optimal surgical visualization. The patient was then placed in the right

lateral decubitus position. Four 5-mm ports were inserted into the left thoracic cavity in the fourth

intercostal space, midaxillary line for the 5-mm 30° scope; fifth intercostal space, anterior-axillary

line; seventh intercostal space, anterior-axillary line; and seventh intercostal space, posterior axillary

line (Fig. 2). Pneumothorax of 4-8 mmHg was established during surgery. After mobilization of the

inferior pulmonary ligament, the endoscope was introduced through the mouth, and the location of

the stenosis was identified. The stenotic site was about 2 cm proximal to the diaphragm. The area of

esophageal stricture was circumferentially dissected with sparing of both vagus nerves and encircled

with vessel tape. With traction of the vessel tape, the esophageal dissection was extended close to the

diaphragm distally and 4 cm proximally from the stenotic site. Several stay sutures were placed at

the cut line of the esophagus, and diamond-shaped partial resection of the esophageal wall was

carried out using scissors with electrocautery (Fig. 3A). The length of the resected stenotic segments

was about 2.0×1.5 cm. After placing a nasogastric tube, a transverse closure was carried out using 5

interrupted 4-0 absorbable sutures (Fig. 3B). Finally, a chest tube was placed through the port site.

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Histologic examination of the resected specimen revealed fibromuscular thickening and fibrosis. A

minor leakage was observed postoperatively for 4 days, but this stopped with conservative treatment.

One month after surgery, balloon dilatation was performed due to slight stenosis at the anastomotic

site, revealing an easily dilatable stenosis. The postoperative esophagogram showed good results

with smooth passage of barium. The patient is asymptomatic and eats normally 5 years after surgery.

1.2 Case 2: 13-month-old boy with CES at the level of T10 due to TBR

Partial esophageal resection and transverse anastomosis followed by anterior partial

fundoplication by a laparoscopic approach were performed for stenosis at the level of T10 (Fig. 1B).

Under general anesthesia, the patient was placed in the supine position. Before surgery, an

esophageal balloon dilator was placed via a nostril into the esophagus to determine the stenotic

segment of the esophagus during surgery. Four 5-mm ports were used in the umbilicus for the 5-mm

30° scope, the right and left epigastric regions, and the left lateral region. Pneumoperitoneum of 10

mmHg was established. A Nathanson retractor was introduced directly at the subxiphoid region to

retract the liver (Fig. 4). First, the esophagus was circumferentially dissected, identifying the

posterior and anterior vagus nerves. The hepatic branch of the vagus nerve was not divided. The

esophagus was encircled with vessel tape above the hepatic branch of the vagus nerve. By traction of

the vessel tape, the hiatus was opened, and further dissection was carried out until enough of the

intra-abdominal part of the esophagus was obtained. The lower esophagus was then mobilized for

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about 5 cm. Next, the balloon was inflated to identify the stenotic site under fluoroscopy. The

stenotic site was about 2.5 cm proximal to the gastroesophageal junction. The anterior vagus nerve

was mobilized from the esophagus and looped to obtain sufficient excision of the stenosis. Several

stay sutures were placed at the cut line of the esophagus. With traction of stay sutures, a

diamond-shaped partial resection of the esophageal wall from the 10- to the 3-o’clock position was

made across the stenotic site using scissors with electrocautery (Fig. 5A). The stenotic site was noted

to be stiff and revealed the presence of cartilage. The length of the resected stenotic segments was

about 2.0×1.5 cm. A nasogastric tube was placed, and the esophagus was then closed transversely

with 6 interrupted 3-0 absorbable sutures (Fig. 5B). The posterior crura were approximated, and the

esophagus was secured to the crura at the 10-, 2-, and 5-o’clock positions. Finally, Thal

fundoplication was performed to reinforce the suture line and prevent gastroesophageal reflux (GER).

Histologic examination of the resected specimen revealed cartilage, tracheobronchial glands, and

ciliated epithelium in the esophageal wall. The patient made an uneventful recovery. One month

after surgery, balloon dilatation was performed due to slight stenosis, revealing an easily dilatable

stenosis. The esophagogram 4 months after surgery showed smooth passage of barium without GER

(Fig. 6). The patient is asymptomatic and eats normally 6 months after surgery.

Discussion

CES is a rare anomaly and divided into 3 types: TBR, FM, and MW. Generally, the symptoms of

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vomiting and regurgitation become significant when the patients start semi-solid and solid foods.

CES commonly affects the lower esophagus, and the diagnosis is made with esophagography and

endoscopy. EUS may be useful to distinguish between TBR and FM by detecting the cartilage [3],

but the precise diagnosis is difficult. In many cases, the diagnosis is made by intraoperative findings

and histologic study of the resected specimen. Appropriate therapeutic strategies, including

endoscopic dilatation and surgical repair, still remain controversial [2][3][4][5][6][7][8]. Generally,

endoscopic dilatation is attempted first. If ineffective, surgical repair is required. Many surgical

procedures, including segmental resection, partial resection, and myectomy of the esophageal wall,

have been reported [2][3][4][5][6][7][13]. The preferred treatment for TBR is resection of the

stenotic segment with end-to-end anastomosis. Conventionally, the surgical approach was performed

via either laparotomy or thoracotomy, depending on the stenotic site. Recently, minimally invasive

surgery using a thoracoscopic or laparoscopic approach has been reported for the treatment of CES

[9][10][11][12]. Deshpande et al [10]. reported laparoscopic esophagoplasty for TBR. In the present

two children with CES, endoscopic esophagoplasty that consisted of a diamond-shaped partial

resection of the esophageal wall and transverse closure was performed. A thoracoscopic or

laparoscopic approach was chosen depending on the stenotic site. In case 1 of CES at the level of T9,

thoracoscopic esophagoplasty was performed. In case 2 of CES at the level of T10, laparoscopic

esophagoplasty followed by anterior partial fundoplication was performed. The postoperative

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esophagogram showed slight stenosis at the anastomotic site in both, but it was easily dilated by

balloon dilatation one month after surgery. Anastomotic leakage, anastomotic stricture, and GER are

postoperative complications. In the laparoscopic approach, a fundoplication should be performed to

prevent not only GER, but also anastomotic leakage. In conclusion, thoracoscopic or laparoscopic

partial resection of the esophageal wall and transverse anastomosis are considered effective, less

invasive, and safe in the treatment of CES, however, the longer follow-up period is necessary

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References

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[3] Takamizawa S, Tsugawa C, Mouri N, Satoh S, Kanegawa K, Nishijima E, et al. Congenital

esophageal stenosis: therapeutic strategy based on etiology. J Pediatr Surg 2002;37:197-201.

[4] Vasudevan SA, Kerendi F, Lee H, Ricketts RR. Management of congenital esophageal stenosis. J

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[9] Martinez-Ferro M, Rubio M, Piaggio L, Laje P. Thoracoscopic approach for congenital

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esophageal stenosis. J Pediatr Surg 2006;41:E5-7.

[10] Deshpande AV, Shun A. Laparoscopic treatment of esophageal stenosis due to tracheobronchial

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[11] van Poll D, van der Zee DC. Thoracoscopic treatment of congenital esophageal stenosis in

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[12] Takazawa S, Uchida H, Kawashima H, Tanaka Y, Sato K, Jimbo T, et al. Successful left

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Consent

Written informed consent was obtained from the patient for publication of this case report and

accompanying images. A copy of the written consent is available for review by the Editor-in-Chief

of this journal on request.

Conflict of interest

The authors have no conflict of interest to disclose.

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Figure Legend

Fig. 1 A. Esophagogram in case 1 showing a stenosis at the level of T9. B. Esophagogram

in case 2 showing a stenosis at the level of T10.

Fig. 2 Port placement in case 1.

Fig. 3 Case 1. A. Diamond-shaped partial resection of the esophageal wall across the

stenotic site (arrow). B. The esophagus was closed transversely with 5 interrupted 4-0

absorbable sutures (arrow head).

Fig. 4 Port placement in case 2.

Fig. 5 Case 2. A. Anterior vagus nerve was mobilized from the esophagus and looped

(arrow). Diamond-shaped partial resection of the esophageal wall from the 10- to 3-o’clock

position was made (arrow head). B. The esophagus was closed transversely with 6 interrupted

3-0 absorbable sutures.

Fig. 6 Esophagogram 4 months after surgery in case 2 showing smooth passage of barium without

gastroesophageal reflux.

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