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Case Study
Video-assisted thoracic surgery for pulmonary sequestration
Asian Cardiovascular & Thoracic Annals 0(0) 1–3 ß The Author(s) 2015 Reprints and permissions: sagepub.co.uk/journalsPermissions.nav DOI: 10.1177/0218492315586480 aan.sagepub.com
Koray Aydog˘du, Ebru Sayılır, Funda I˙ncekara, Go¨ktu¨rk Fındık and Sadi Kaya
Abstract Bronchopulmonary sequestration is a rare developmental abnormality. Most cases are asymptomatic and found incidentally. The definitive treatment for bronchopulmonary sequestration is surgical excision. An 18-year-old man was admitted to our clinic with longstanding cough, fever, and dense sputum. Chest computed tomography identified cystic bronchiectasis in common areas of the left lower lobe, and parenchymal destruction with air-fluid levels. A left lower lobectomy was performed via a video-thoracoscopic approach.
Keywords Bronchopulmonary sequestration, lobectomy, video-assisted thoracic surgery
Introduction Bronchopulmonary sequestration (BPS) is a congenital anomaly consisting of a mass of nonfunctioning lung tissue. There is a lack of normal communication with the tracheobronchial tree and it receives its arterial blood supply from the systemic circulation, commonly from the aorta. Surgical resection is the recommended treatment for BPS. Surgery is generally performed via thoracotomy, but thoracoscopic resection can be performed for BPS.
Case report An 18-year-old man was admitted to our clinic with cough, fever, and dense sputum. His complaints were of long duration and he often received medical treatment in health centers, but he was never fully healed. On physical examination, there were rough breath sounds and rhonchi at the lower zone of the left lung. Chest radiography showed a prominent bronchovascular structure in the left lower zone (Figure 1A). Laboratory tests were normal except for free triiodothyronine near the top of the normal range in thyroid function tests. There were no endobronchial lesions observed on fiberoptic bronchoscopy. Chest computed tomography identified cystic bronchiectasis in common areas of the left lower lobe, and areas of parenchymal destruction with air-fluid levels (Figure 1B). We decided
to perform a video-thoracoscopic left lower lobectomy with the diagnosis of cystic bronchiectasis and sequestration. The thoracoscopic approach follows the same principles as the open technique via thoracotomy. The patient underwent double-lumen intubation and was placed in the lateral decubitus position. We made a utility incision and 2 port incisions. The first port was placed in the 7th or 8th intercostal space for the camera. After observation of the thoracic cavity, the utility incision was made the 4th intercostal space. The 3rd incision was for other instruments. During the operation, the pulsatile vascular structures arising directly from the distal aorta were in close proximity to the diaphragm. We carefully dissected the pulmonary ligament for dissection of the left inferior pulmonary vein, considering the possibility of sequestration (Figure 2A). The connection between the vascular structure and the lower lobe was exposed and cut with a vascular stapler (Figure 2B). The lobectomy was smoothly completed with inferior pulmonary Department of Thoracic Surgery, Atatu¨rk Chest Diseases and Thoracic Surgery Research and Training Hospital, Ankara, Turkey Corresponding author: Koray Aydog˘du, Department of Thoracic Surgery, Atatu¨rk Chest Diseases and Thoracic Surgery Research and Training Hospital, Ankara, Turkey. Email: [email protected]
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Figure 1. (A) Preoperative chest radiograph showing a prominent bronchovascular structure in the left lower zone. (B) Chest computed tomography identified cystic bronchiectasis in common areas of the left lower lobe and areas of parenchymal destruction with air-fluid levels.
Figure 2. (A) The pulsatile vascular structure directly from the distal aorta, in close proximity to the diaphragm. (B) The vascular structure were cut with a vascular stapler.
vein, common basal artery, and bronchial dissection. Postoperative pathology reported lung parenchyma compatible with sequestration. The drains were removed on the 3rd postoperative day, and the patient was discharged without any problem. At the 6-month follow-up, the patient reported no further problems (Figure 3).
Discussion BPS is a rare developmental abnormality accounting for 0.15%–6.4% of all congenital lung anomalies.1 BPS is classified anatomically into two groups: intralobar pulmonary sequestration and extralobar pulmonary sequestration. The most common form of BPS is intralobar pulmonary sequestration which accounts for 75% of BPS. BPS is generally located in the lower lobes, especially the left lower lobe as in our case.2–4 In both types of BPS, systemic arterial supply is usually from the thoracic or abdominal aorta, but the venous drainage is to the systemic circulation in extralobar pulmonary sequestration and to the pulmonary vein in
intralobar pulmonary sequestration.4 In our case, systemic arterial supply was from the thoracic aorta and the venous drainage was to the pulmonary vein. Most BPS cases are asymptomatic and found incidentally on routine radiography. The initial symptoms often appear in young patients, including recurrent cough, sputum, fever, chest pain, and hemoptysis. Our patient had recurrent cough, sputum, and fever continuing for a long time. BPS is often misdiagnosed as a congenital pulmonary cyst or bronchiectasis complicated by infection, with a preoperative misdiagnosis rate of 71%.1,5,6 The most useful diagnostic methods for pulmonary sequestration are computed tomography angiography, magnetic resonance angiography, and digital subtraction angiography, which may show the aberrant arterial supply.7 The parenchymal abnormalities associated with BPS are best visualized with computed tomography, although the appearance is variable. The most common finding is a solid mass. The lesion may be homogeneous or heterogeneous and may include cystic changes. A large cavitary lesion with an air-fluid level, a collection of many
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Figure 3. (A) The healed utility incision and two port incisions. (B) Postoperative chest radiograph.
small cystic lesions containing air or fluid, or a welldefined cystic mass are rare findings. Digital subtraction angiography is the gold standard for preoperative diagnosis.1,8,9 The definitive treatment for BPS is surgical excision. Once a patient is diagnosed with pulmonary sequestration, resection is suggested regardless of symptoms. In the past, a posterolateral thoracotomy was the main approach for pulmonary sequestration because of the hidden aberrant arteries, pleural adhesions, and other reasons.1 One of the most important aspects of resection is careful identification of vascular structures because the arterial supply to a BPS may arise from the subdiaphragmatic abdominal aorta. In our case, with the help of thoracoscopy, the inferior pulmonary ligament was carefully dissected and a pulsatile vascular structure (artery) originating directly from the descending aorta close to the diaphragm was identified. The diameter of artery was 12 mm. We used a endoscopic vascular stapler after placing a vascular clamp around the artery to prevent uncontrollable bleeding. The vascular clamp was opened carefully because we were cautious about the application of a vascular stapler to such a large systemic artery originating directly from the aorta. With our experienced surgical team, thoracoscopic separation of the pleural adhesions and dissection of the pulmonary ligament was not as difficult as expected. Moreover, thoracoscopy helped to decrease recovery time and lessen postoperative pain. Thus video-assisted lobectomy for pulmonary sequestration can be recommended as an alternative to the conventional thoracotomy approach. Funding This research received no specific grant from any funding agency in the public, commercial, or not-for-profit sectors.
Conflict of interest statement None declared.
References 1. Shen JF, Zhang XX and Li SB. Complete video-assisted thoracoscopic surgery for pulmonary sequestration. J Thorac Dis 2013; 5: 31–315. 2. Van Raemdonck D, De Boeck K, Devlieger H, et al. Pulmonary sequestration: a comparison between pediatric and adult patients. Eur J Cardiothorac Surg 2001; 19: 388–395. 3. Wei Y and Li F. Pulmonary sequestration: a retrospective analysis of 2625 cases in China. Eur J Cardiothorac Surg 2011; 40: 39–42. 4. Ersin Arslan. Pulmonary sequestration with different clinical and radiological findings. Tu¨rk Go¨g˘u¨s Kalp Damar Cerrahisi Dergisi 2013; 21: 110–113. Available at: http:// tgkdc.dergisi.org/pdf/pdf_TGKDC_1724.pdf. Accessed April 20, 2015. 5. Matsuoka H and Nohara H. Pulmonary sequestration with high levels of tumor markers tending to be misdiagnosed as lung cancer. Jpn J Thorac Cardiovasc Surg 2006; 54: 117–119. 6. Ahmed M, Jacobi V and Vogl TJ. Multislice CT and CT angiography for non-invasive evaluation of bronchopulmonary sequestration. Eur Radiol 2004; 14: 2141–2143. 7. Lin CH, Chuang CY, Hsia JY, Lee MC, Shai SE, Yang SS and Hsu CP. Pulmonary sequestration-differences in diagnosis and treatment in a single institution. J Chin Med Assoc 2013; 76: 385–389. 8. Ko SF, Ng SH, Lee TY, et al. Noninvasive imaging of bronchopulmonary sequestration. AJR Am J Roentgenol 2000; 175: 1005–1012. 9. Walker CM, Wu CC, Gilman MD, Godwin JD 2nd, Shepard JA and Abbott GF. The imaging spectrum of bronchopulmonary sequestration. Curr Probl Diagn Radiol 2014; 43: 100–114.
Downloaded from aan.sagepub.com at HEC Montreal on June 21, 2015
(AAN)
[1–3] [PREPRINTER stage]
Case Study
Video-assisted thoracic surgery for pulmonary sequestration
Asian Cardiovascular & Thoracic Annals 0(0) 1–3 ß The Author(s) 2015 Reprints and permissions: sagepub.co.uk/journalsPermissions.nav DOI: 10.1177/0218492315586480 aan.sagepub.com
Koray Aydog˘du, Ebru Sayılır, Funda I˙ncekara, Go¨ktu¨rk Fındık and Sadi Kaya
Abstract Bronchopulmonary sequestration is a rare developmental abnormality. Most cases are asymptomatic and found incidentally. The definitive treatment for bronchopulmonary sequestration is surgical excision. An 18-year-old man was admitted to our clinic with longstanding cough, fever, and dense sputum. Chest computed tomography identified cystic bronchiectasis in common areas of the left lower lobe, and parenchymal destruction with air-fluid levels. A left lower lobectomy was performed via a video-thoracoscopic approach.
Keywords Bronchopulmonary sequestration, lobectomy, video-assisted thoracic surgery
Introduction Bronchopulmonary sequestration (BPS) is a congenital anomaly consisting of a mass of nonfunctioning lung tissue. There is a lack of normal communication with the tracheobronchial tree and it receives its arterial blood supply from the systemic circulation, commonly from the aorta. Surgical resection is the recommended treatment for BPS. Surgery is generally performed via thoracotomy, but thoracoscopic resection can be performed for BPS.
Case report An 18-year-old man was admitted to our clinic with cough, fever, and dense sputum. His complaints were of long duration and he often received medical treatment in health centers, but he was never fully healed. On physical examination, there were rough breath sounds and rhonchi at the lower zone of the left lung. Chest radiography showed a prominent bronchovascular structure in the left lower zone (Figure 1A). Laboratory tests were normal except for free triiodothyronine near the top of the normal range in thyroid function tests. There were no endobronchial lesions observed on fiberoptic bronchoscopy. Chest computed tomography identified cystic bronchiectasis in common areas of the left lower lobe, and areas of parenchymal destruction with air-fluid levels (Figure 1B). We decided
to perform a video-thoracoscopic left lower lobectomy with the diagnosis of cystic bronchiectasis and sequestration. The thoracoscopic approach follows the same principles as the open technique via thoracotomy. The patient underwent double-lumen intubation and was placed in the lateral decubitus position. We made a utility incision and 2 port incisions. The first port was placed in the 7th or 8th intercostal space for the camera. After observation of the thoracic cavity, the utility incision was made the 4th intercostal space. The 3rd incision was for other instruments. During the operation, the pulsatile vascular structures arising directly from the distal aorta were in close proximity to the diaphragm. We carefully dissected the pulmonary ligament for dissection of the left inferior pulmonary vein, considering the possibility of sequestration (Figure 2A). The connection between the vascular structure and the lower lobe was exposed and cut with a vascular stapler (Figure 2B). The lobectomy was smoothly completed with inferior pulmonary Department of Thoracic Surgery, Atatu¨rk Chest Diseases and Thoracic Surgery Research and Training Hospital, Ankara, Turkey Corresponding author: Koray Aydog˘du, Department of Thoracic Surgery, Atatu¨rk Chest Diseases and Thoracic Surgery Research and Training Hospital, Ankara, Turkey. Email: [email protected]
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Figure 1. (A) Preoperative chest radiograph showing a prominent bronchovascular structure in the left lower zone. (B) Chest computed tomography identified cystic bronchiectasis in common areas of the left lower lobe and areas of parenchymal destruction with air-fluid levels.
Figure 2. (A) The pulsatile vascular structure directly from the distal aorta, in close proximity to the diaphragm. (B) The vascular structure were cut with a vascular stapler.
vein, common basal artery, and bronchial dissection. Postoperative pathology reported lung parenchyma compatible with sequestration. The drains were removed on the 3rd postoperative day, and the patient was discharged without any problem. At the 6-month follow-up, the patient reported no further problems (Figure 3).
Discussion BPS is a rare developmental abnormality accounting for 0.15%–6.4% of all congenital lung anomalies.1 BPS is classified anatomically into two groups: intralobar pulmonary sequestration and extralobar pulmonary sequestration. The most common form of BPS is intralobar pulmonary sequestration which accounts for 75% of BPS. BPS is generally located in the lower lobes, especially the left lower lobe as in our case.2–4 In both types of BPS, systemic arterial supply is usually from the thoracic or abdominal aorta, but the venous drainage is to the systemic circulation in extralobar pulmonary sequestration and to the pulmonary vein in
intralobar pulmonary sequestration.4 In our case, systemic arterial supply was from the thoracic aorta and the venous drainage was to the pulmonary vein. Most BPS cases are asymptomatic and found incidentally on routine radiography. The initial symptoms often appear in young patients, including recurrent cough, sputum, fever, chest pain, and hemoptysis. Our patient had recurrent cough, sputum, and fever continuing for a long time. BPS is often misdiagnosed as a congenital pulmonary cyst or bronchiectasis complicated by infection, with a preoperative misdiagnosis rate of 71%.1,5,6 The most useful diagnostic methods for pulmonary sequestration are computed tomography angiography, magnetic resonance angiography, and digital subtraction angiography, which may show the aberrant arterial supply.7 The parenchymal abnormalities associated with BPS are best visualized with computed tomography, although the appearance is variable. The most common finding is a solid mass. The lesion may be homogeneous or heterogeneous and may include cystic changes. A large cavitary lesion with an air-fluid level, a collection of many
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Figure 3. (A) The healed utility incision and two port incisions. (B) Postoperative chest radiograph.
small cystic lesions containing air or fluid, or a welldefined cystic mass are rare findings. Digital subtraction angiography is the gold standard for preoperative diagnosis.1,8,9 The definitive treatment for BPS is surgical excision. Once a patient is diagnosed with pulmonary sequestration, resection is suggested regardless of symptoms. In the past, a posterolateral thoracotomy was the main approach for pulmonary sequestration because of the hidden aberrant arteries, pleural adhesions, and other reasons.1 One of the most important aspects of resection is careful identification of vascular structures because the arterial supply to a BPS may arise from the subdiaphragmatic abdominal aorta. In our case, with the help of thoracoscopy, the inferior pulmonary ligament was carefully dissected and a pulsatile vascular structure (artery) originating directly from the descending aorta close to the diaphragm was identified. The diameter of artery was 12 mm. We used a endoscopic vascular stapler after placing a vascular clamp around the artery to prevent uncontrollable bleeding. The vascular clamp was opened carefully because we were cautious about the application of a vascular stapler to such a large systemic artery originating directly from the aorta. With our experienced surgical team, thoracoscopic separation of the pleural adhesions and dissection of the pulmonary ligament was not as difficult as expected. Moreover, thoracoscopy helped to decrease recovery time and lessen postoperative pain. Thus video-assisted lobectomy for pulmonary sequestration can be recommended as an alternative to the conventional thoracotomy approach. Funding This research received no specific grant from any funding agency in the public, commercial, or not-for-profit sectors.
Conflict of interest statement None declared.
References 1. Shen JF, Zhang XX and Li SB. Complete video-assisted thoracoscopic surgery for pulmonary sequestration. J Thorac Dis 2013; 5: 31–315. 2. Van Raemdonck D, De Boeck K, Devlieger H, et al. Pulmonary sequestration: a comparison between pediatric and adult patients. Eur J Cardiothorac Surg 2001; 19: 388–395. 3. Wei Y and Li F. Pulmonary sequestration: a retrospective analysis of 2625 cases in China. Eur J Cardiothorac Surg 2011; 40: 39–42. 4. Ersin Arslan. Pulmonary sequestration with different clinical and radiological findings. Tu¨rk Go¨g˘u¨s Kalp Damar Cerrahisi Dergisi 2013; 21: 110–113. Available at: http:// tgkdc.dergisi.org/pdf/pdf_TGKDC_1724.pdf. Accessed April 20, 2015. 5. Matsuoka H and Nohara H. Pulmonary sequestration with high levels of tumor markers tending to be misdiagnosed as lung cancer. Jpn J Thorac Cardiovasc Surg 2006; 54: 117–119. 6. Ahmed M, Jacobi V and Vogl TJ. Multislice CT and CT angiography for non-invasive evaluation of bronchopulmonary sequestration. Eur Radiol 2004; 14: 2141–2143. 7. Lin CH, Chuang CY, Hsia JY, Lee MC, Shai SE, Yang SS and Hsu CP. Pulmonary sequestration-differences in diagnosis and treatment in a single institution. J Chin Med Assoc 2013; 76: 385–389. 8. Ko SF, Ng SH, Lee TY, et al. Noninvasive imaging of bronchopulmonary sequestration. AJR Am J Roentgenol 2000; 175: 1005–1012. 9. Walker CM, Wu CC, Gilman MD, Godwin JD 2nd, Shepard JA and Abbott GF. The imaging spectrum of bronchopulmonary sequestration. Curr Probl Diagn Radiol 2014; 43: 100–114.
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