Ann Thorac Surg 2005;80:1266-1269
© 2005 The Society of Thoracic Surgeons
Original article: General thoracic
Video-Assisted Thoracoscopic Surgery for Pulmonary Sequestration in Children
Pascal de Lagausie, MD, PhD
a
,
*
,
Arnaud Bonnard, MD
c
,
Dominique Berrebi, MD, PhD
d
,
Philippe Petit, MD
b
,
Sophie Dorgeret, MD
e
,
Jean Michel Guys, MD
a
a Department of Pediatric Surgery, Hôpital la Timone, Marseille, France
b Department of Pediatric Radiology, Hôpital la Timone, Marseille, France
c Department of Pediatric Surgery, Hôpital Robert Debré, Paris, France
d Department of Anatomopathology, Hôpital Robert Debré, Paris, France
e Department of Pediatric Radiology, Hôpital Robert Debré, Paris, France
Accepted for publication February 1, 2005.
* Address reprint requests to Dr de Lagausie, Department of Pediatric Surgery, Hôpital la Timone, 264 rue Saint Pierre, 13385 Marseille cedex 05, France (Email: pascal.delagausie{at}ap-hm.fr).
 |
Abstract
|
|---|
BACKGROUND: The purpose of this report is to describe our experience with video-assisted thoracoscopic surgery for pulmonary sequestration in children.
METHODS: From May 2001 to June 2004, video-assisted thoracoscopic surgery was attempted for antenatally diagnosed pulmonary sequestration in 8 consecutive infants. Mean age at the time of surgery was 10 months (range, 4 to 44 months). Six lesions were located in the left lower lobe and two in the right lower lobe. Endovascular embolization was attempted before video-assisted thoracoscopic surgery in only 1 patient. All procedures were performed in the lateral decubitus position, and single-lung ventilation was used in all cases.
RESULTS: Conversion to open surgery was necessary in two cases. Video-assisted thoracoscopic surgery was successful in 6 patients. After being identified and isolated, the aberrant artery was controlled by endoscopic ligation, and lobectomy, wedge resection, or sequestration was performed depending on the type of lesion. Mean operative time was 155 minutes. Average hospital stay was 3.5 days. There were no postoperative complications. Follow-up ranged from 4 to 50 months.
CONCLUSIONS: Video-assisted thoracoscopic surgery is technically feasible for pulmonary sequestration. Early resection obviates the risk of infection. Elective ligation of the aberrant artery is a safe alternative to the use of stapling devices or clips. Cosmetic results are excellent.
 |
Introduction
|
|---|
Pulmonary sequestration is a congenital anomaly of lung parenchyma characterized by partial or complete separation of a portion of a lobe without appropriate bronchial and vascular connections [1]. It accounts between 0.15% and 6.45% of all pulmonary malformations [2]. Pulmonary sequestration is classified as either extralobar or intralobar. Extralobar sequestration (ELS), which account for 25% of cases, has its own pleural investment. Intralobar sequestration (ILS) is surrounded by normal lung tissue. Both ELS and ILS receive their blood supply from anomalous systemic arteries, usually arising from the descending aorta. Venous drainage is usually by the pulmonary veins for ILS and by the systemic venous system for ELS [3]. Diagnosis is usually made antenatally. Because of the risk of infection [4], the policy at our institution is to perform resection within the first year of life. Previous studies have shown that video-assisted thoracoscopic surgery (VATS) is feasible without higher morbidity than open surgery [4, 5]. The purpose of this report is to describe our experience with thoracoscopic surgery with special emphasis on management of the aberrant artery.
 |
Patients and Methods
|
|---|
From May 2001 to June 2004, VATS was attempted for pulmonary sequestration in 8 consecutive infants (Table 1). In all cases the lower thoracic mass, including two on the right and six on the left, was identified antenatally by routine prenatal ultrasound. Diagnosis of pulmonary sequestration was confirmed postnatally by computed tomographic scan in six cases or magnetic resonance imaging in three cases. The feeding artery originated from the descending aorta in all cases. No associated cardiac or diaphragmatic anomalies were noted. Mean age at the time of surgery was 10 months (range, 4 to 44 months), and mean weight was 6.8 kg (range, 4.5 to 12 kg). In one case, a young 44-month-old girl was referred to our institution after two failed embolization attempts.
All VATS procedures were performed with the patient in the lateral decubitus position with single-lung ventilation achieved by occluding the left or right bronchus using a Fogarty balloon catheter placed with flexible bronchoscopic guidance (Fig 1). The surgeon and assistant stood facing the child's back. A single monitor was placed in front of the operating surgeon. Three to five endoscopic ports ranging from 5 to 10 mm (one 10 mm for telescope) were used. Because the EndoGIA (USSC, Autosuture, Tyco Healthcare, Norwalk, CT) is too large for the thoracic cavity of a small child, stapling could not be used. Instead, the systemic artery was occluded by triple ligation using absorbable suture and endocorporeal knots. Clips were never used (Fig 2). Vein ligation was also performed using absorbable suture. For ILS, lobectomy or wedge resection was performed using bipolar cautery or harmonic scalpel (Ethicon, Somerville, NJ). For ELS, sequestrectomy was performed in all cases. The lower trocar site incision was extended sufficiently to allow removal of the specimen. In all cases a chest tube was left in place for 1 to 3 days.

View larger version (114K):
[in this window]
[in a new window]
|
Fig 1. The external end of the Fogarty catheter is fixed to the tracheal tube by using adhesive plasters (Steristrip, 3M, Cergy-Pontoise, France). Fogarty balloon is placed in the main bronchus (left in this case) under flexible bronchoscopic control.
|
|

View larger version (143K):
[in this window]
[in a new window]
|
Fig 2. Control of systemic artery in one case of left lower extralobar sequestration. Dissection (A), triple ligation (B), section (C), and monitoring the absence of bleeding (D).
|
|
 |
Results
|
|---|
Two children required conversion to open surgery at the beginning of our experience because of failure to obtain sufficient exposure in one case and lack of a fissure in another case involving ILS. Video-assisted thoracoscopic surgery was successful in 6 children. The aberrant artery was dissected as far from the sequestrated lung as possible on the descending aorta (near diaphragm) or thoracic aorta and controlled by triple ligation. Bleeding did not occur, and no child required transfusion. The mean duration of the six successful procedures was 120 minutes (range, 80 to 190 minutes). Histologic examination demonstrated ELS in three cases and ILS in five cases including one associated with a cystic adenomatoid malformation. Mean postoperative analgesic time (acetaminophen and morphine analgesics) was 4.1 days (range, 3 to 6 days). Mean duration of hospitalization calculated including the 2 patients requiring open surgery was 4.6 days (range, 3 to 8 days). The only postoperative complication was pneumonia treated with antibiotic therapy and physiotherapy. Follow-up ranged from 4 to 50 months. Cosmetic results ascertained by visual inspection on a postoperative follow-up examination were excellent (Fig 3).
 |
Comment
|
|---|
Cystic lung lesions including pulmonary sequestration and congenital cystic adenomatoid malformation are the most common antenatally diagnosed intrathoracic masses in fetuses. Occurrence of sequestration with congenital cystic adenomatoid malformation has been reported previously [6] and was observed in one case in our series. Children with asymptomatic sequestration are at risk for pulmonary infection and abscess, with greater than half of these complications occurring within the first year of life [4]. Development of malignant epithelial and mesenchymal tumors has been reported in association with congenital cystic adenomatoid malformation [7]. Early surgical intervention provides definitive treatment for pulmonary sequestration and is, in our opinion, recommended.
Minimally invasive techniques have been used to treat a variety of pulmonary lesions. The scope of these techniques has progressed from limited exploration, biopsy, and debridement to extensive technically demanding resection and reconstruction procedures. Video-assisted thoracoscopic surgery now offers surgeons and patients an alternative to open surgery for both ILS and ELS. After a preliminary case report describing VATS for pulmonary sequestration [8], the present report describes results in our initial series.
Development of small instruments and strategies for single-lung ventilation has enabled VATS in small children. In cases involving sequestration, thoracoscopic wedge resection or lobectomy can be performed safely provided that the aberrant feeding artery can be controlled. Thoracoscopic surgery has been shown to cause no greater short-term and long-term morbidity than thoracotomy [4, 9] and has been associated with scoliosis, shoulder muscle girdle deformity, and chest wall deformity [10].
Watine and associates [11] reported postoperative hemothorax in 15% of patients undergoing surgical treatment for pulmonary sequestration and suggested that this complication was caused by inappropriate dissection of the aberrant artery. In this regard it must be emphasized that the aberrant artery can be inflammatory and thus easily damaged [12]. In adults a vascular stapler is often used to section and ligate [13, 14]. Inasmuch as introduction and operation of an EndoGIA requires a 12-mm port and at least 4 cm of intrathoracic space, stapling is difficult or impossible in infants and neonates [9]. In our experience this problem was resolved by dissecting as far from the sequestered lung as possible and performing a triple ligature at the aortic orifice or at the orifice of the thoracic cavity. Clips were never used because of the risk of slipping in the event of intraabdominal retraction of the aberrant artery.
Albanese and colleagues [4] reported an alternative solution for controlling the aberrant artery using the Ligasure vessel sealing system (Valleylab, Boulder, CO), which allows safe and effective sealing vessels up to 7 mm diameter. Their findings showed that this system was ideal for the low-pressure pulmonary circulation in small children. We have not used that system, but our experience indicates that aberrant arteries can sometimes be large with high arterial pressure.
This series confirms that VATS for resection of pulmonary sequestration is feasible and safe in small children. It can be used successfully even in case of unsuccessful preoperative embolization. Endoligation is a reliable technique for controlling the aberrant artery. Safe performance of this technique requires a sound understanding of the regional anatomy and a clear vision of associated three-dimensional relationships.
 |
Notice From the American Board of Thoracic Surgery Regarding Trainees and Candidates for Certification Who Are Called to Military Service Related to the War on Terrorism
|
|---|
The Board appreciates the concern of those who have received emergency calls to military service. They may be assured that the Board will exercise the same sympathetic consideration as was given to candidates in recognition of their special contributions to their country during the Vietnam conflict and the Persian Gulf conflict with regard to applications, examinations, and interruption of training. If you have any questions about how this might affect you, please call the Board office at (312) 202-5900.
Timothy J. Gardner, MD Chairman
The American Board of Thoracic Surgery
 |
References
|
|---|
- de Lagausie P, Van Den Abbeele T, Elmaleh M, Ferkadji L, Maintenant J, Aigrain Y. Bronchial trifurcation in a congenital pulmonary venolobar syndrome Pediatr Pulmonol 2001;31:303-305.[Medline]
- Halkic N, Cuénoud PF, Corthésy ME, Ksontini R, Boumghar M. Pulmonary sequestrationa review of 26 cases. Eur J Cardiothoracic Surg 1998;14:127-133.
- Clements BS, Warner JO. Pulmonary sequestration and related congenital broncho-pulmonary malformationsnomenclature and classification based on anatomical and embryologic considerations. Thorax 1987;42:401-408.[Abstract/Free Full Text]
- Albanese CT, Sydorak RM, Tsao K, Lee H. Thoracoscopic lobectomy for prenatally diagnosed lung lesions J Pediatr Surg 2003;38:553-555.[Medline]
- Mezzetti M, Dell'Agnola CA, Bedoni M, Cappelli R, Fumagalli F, Panigalli T. Video-assisted thoracoscopic resection of pulmonary sequestration in an infant Ann Thorac Surg 1996;61:1836-1838.[Abstract/Free Full Text]
- Gluer S, Scharf A, Ure BM. Thoracoscopic resection of extralobar sequestration in a neonate J Pediatr Surg 2002;37:1629-1631.[Medline]
- Granata C, Gambini C, Balducci T, et al. Bronchioalveolar carcinoma arising in congenital cystic adenomatoid malformation in a childa case report and review on malignancies originating in congenital cystic adenomatoid malformation. Pediatr Pulmonol 1998;25:62-66.[Medline]
- Bonnard A, Malbezin S, Ferkdadji L, Luton D, Aigrain Y, de Lagausie P. Pulmonary sequestration in childrenis the thoracoscopic approach a good option?. Surg Endosc 2004;18:80-82.[Medline]
- Rothenberg SS. Experience with thoracoscopic lobectomy in infants and children J Pediatr Surg 2003;38:102-104.[Medline]
- Bal S, Eishershari H, Celiker R, Celiker A. Thoracic sequels after thoracotomies in children with congenital cardiac disease Cardiol Young 2003;13:264-267.[Medline]
- Watine O, Mensier E, Delecluse P, Ribet M. Pulmonary sequestration treated by video thoracoscopic resection J Pediatr Surg 2002;37:1629-1631.
- Tanaka T, Ueda K, Sakano H, Hayashi M, Li TS, Zempo N. Video-assisted thoracoscopic surgery for intralobar pulmonary sequestration Surgery 2003;133:216-218.[Medline]
- Wan IYP, Lee TW, Sihoe ADL, Yim APC. Video-assisted thoracoscopic surgery resection for pulmonary sequestration Ann Thorac Surg 2002;73:639-640.[Abstract/Free Full Text]
- Klena JW, Daneck SJ, Bostwick TK, Romero M, Johnson JA. Video-assisted thoracoscopic resection for intralobar sequestrationsingle modality treatment with video-assisted thoracic surgery. J Thorac Cardiovasc Surg 2003;126:857-859.[Free Full Text]
This article has been cited by other articles:

|
 |

|
 |
 
S. Gezer, I. Tastepe, M. Sirmali, G. Findik, H. Turut, S. Kaya, N. Karaoglanoglu, and G. Cetin
Pulmonary sequestration: A single-institutional series composed of 27 cases
J. Thorac. Cardiovasc. Surg.,
April 1, 2007;
133(4):
955 - 959.
[Abstract]
[Full Text]
[PDF]
|
 |
|

|
 |

|
 |
 
T. Suda, S. Hasegawa, K. Negi, and Y. Hattori
Video-assisted thoracoscopic surgery for extralobar pulmonary sequestration.
J. Thorac. Cardiovasc. Surg.,
September 1, 2006;
132(3):
707 - 708.
[Full Text]
[PDF]
|
 |
|

|
 |

|
 |
 
C. R. Morse, M. B. Ishitani, and S. D. Cassivi
Video-assisted resection of bilateral intralobar pulmonary sequestrations
J. Thorac. Cardiovasc. Surg.,
April 1, 2006;
131(4):
917 - 918.
[Full Text]
[PDF]
|
 |
|