Thoracoscopic Lobectomy for Synchronous Intralobar Pulmonary Sequestration and Lung Cancer
2013; Elsevier BV; Volume: 96; Issue: 2 Linguagem: Inglês
10.1016/j.athoracsur.2012.12.063
ISSN1552-6259
AutoresTom Kai Ming Wang, T. Oh, Tharumenthiran Ramanathan,
Tópico(s)Tracheal and airway disorders
ResumoBronchopulmonary sequestration is a rare congenital pulmonary malformation for which surgical resection is recommended, and several reports have described successful resection by video-assisted thoracoscopic surgery. Coexistence of sequestration with lung malignancy is extremely rare. We report the first case of thoracoscopic resection of synchronous intralobar pulmonary sequestration and non-small cell lung cancer. Bronchopulmonary sequestration is a rare congenital pulmonary malformation for which surgical resection is recommended, and several reports have described successful resection by video-assisted thoracoscopic surgery. Coexistence of sequestration with lung malignancy is extremely rare. We report the first case of thoracoscopic resection of synchronous intralobar pulmonary sequestration and non-small cell lung cancer. Bronchopulmonary sequestration makes up 0.15% to 6.4% of congenital pulmonary anomalies and is defined as an area of lung parenchyma not connected with the tracheobronchial system with its own aberrant systemic arterial supply, usually from the aorta [1Savic B. Birtel F.J. Tholen W. Funke H.D. Knoche R. Lung sequestration: report of seven cases and review of 540 published cases.Thorax. 1979; 34: 96-101Crossref PubMed Scopus (517) Google Scholar]. The coexistence of lung malignancy and bronchopulmonary sequestration is extremely rare [2Lawal L. Mikroulis D. Eleftheriadis S. Karros P. Bougioukas I. Bougioukas G. Adenocarcinoma in pulmonary sequestration.Asian Cardiovasc Thorac Ann. 2011; 19: 433-435Crossref PubMed Scopus (15) Google Scholar]. Thoracoscopic lobectomy for lung cancer is well established and thoracoscopic resection of bronchopulmonary sequestration has been previously reported [3de Lagausie P. Bonnard A. Berrebi D. Petit P. Dorgeret S. Guys J.M. Video-assisted thoracoscopic surgery for pulmonary sequestration in children.Ann Thorac Surg. 2005; 80: 1266-1269Abstract Full Text Full Text PDF PubMed Scopus (39) Google Scholar, 4Tsang F.H. Chung S.S. Sihoe A.D. Video-assisted thoracic surgery for bronchopulmonary sequestration.Interact Cardiovasc Thorac Surg. 2006; 5: 424-426Crossref PubMed Scopus (28) Google Scholar, 5Kestenholz P.B. Schneiter D. Hillinger S. Lardinois D. Weder W. Thoracoscopic treatment of pulmonary sequestration.Eur J Cardiothorac Surg. 2006; 29: 815-818Crossref PubMed Scopus (79) Google Scholar].A 65-year-old man was incidentally found (on magnetic resonance imaging) to have a 5.5 × 3.5 cm cystic mass in the left hemithorax. Subsequent chest radiograph showed a left retrocardiac opacity and a computed tomography (CT) showed the presence of either a neuroenteric cyst due to its paraspinal location, or a bronchopulmonary sequestration. Follow-up CT demonstrated a new 2.5-cm spiculated nodule in the left lower lobe just medial to the cystic lesion and an arterial connection to the cystic lesion from the descending thoracic aorta just superior to the diaphragm, which provided convincing evidence of intralobar pulmonary sequestration (Fig 1). A CT-guided biopsy of the lung nodule established a histologic diagnosis of adenocarcinoma. Positron emission tomography confirmed stage 1A disease (T1BN0M0). Preoperative pulmonary function tests were within normal limits and the decision was made to proceed to resection of both lesions by video-assisted thoracoscopic surgery (VATS) left lower lobectomy.Three ports and a utility incision were made. After confirmation of the site of the sequestration and lung primary, the left lower lobe was retracted cephalad. The inferior pulmonary ligament was identified and the aberrant arterial supply to the sequestration noted (Fig 2). Careful dissection of the aberrant vessel was performed and a silk tie passed around it. The vessel was divided with an Endo-GIA vascular stapler (Covidien, Norwalk, CT). A Chitwood-DeBakey straight aortic cross clamp was positioned proximally on the aberrant vessel in the event of stapler misadventure. A “fissure-less” technique was used to complete the left lower lobectomy with sequential division using endoscopic staplers of the inferior pulmonary vein, the lower lobe bronchus, and the continuation of the pulmonary artery to the lower lobe. After the fissure was completed, the lobe was placed in a bag and removed through the utility incision. Operative time was 105 minutes. Blood loss was less than 50 mL and the patient made a routine uncomplicated recovery; he was discharged home on postoperative day 2. Figure 3 shows the resected lung specimen with the tumor and sequestration.Fig 2Thoracoscopic image of aberrant artery (arrow A) in the inferior pulmonary ligament supplying the bronchopulmonary sequestration.View Large Image Figure ViewerDownload (PPT)Fig 3Specimen image of excised lung tissue with (arrow T) lung tumor and (arrow S) pulmonary sequestration.View Large Image Figure ViewerDownload (PPT)CommentBronchopulmonary sequestration is classified anatomically as intralobar (∼75%) or extralobar (∼25%), depending on whether the sequestrated tissue shares the same pleural investment with normal lung tissue [1Savic B. Birtel F.J. Tholen W. Funke H.D. Knoche R. Lung sequestration: report of seven cases and review of 540 published cases.Thorax. 1979; 34: 96-101Crossref PubMed Scopus (517) Google Scholar]. It has a systemic aberrant arterial supply from the thoracic aorta 74% of the time, more than 1 aberrant artery in 15%, while venous return is to the pulmonary veins in 96%. Cystic changes, fibrosis and chronic inflammation of lung parenchyma are the main histologic features [6Stocker J.T. Sequestrations of the lung.Semin Diagn Pathol. 1986; 2: 106-121Google Scholar]. Clinical presentation can range from cough, hemoptysis, pneumonia, and congestive heart failure to potential associations with other pulmonary and skeletal anomalies such as congenital diaphragmatic hernia and scoliosis [1Savic B. Birtel F.J. Tholen W. Funke H.D. Knoche R. Lung sequestration: report of seven cases and review of 540 published cases.Thorax. 1979; 34: 96-101Crossref PubMed Scopus (517) Google Scholar, 7Abbey P. Das C.J. Pangtey G.S. Seith A. Dutta R. Kumar A. Imaging in bronchopulmonary sequestration.J Med Imaging Radiat Oncol. 2009; 53: 22-31Crossref PubMed Scopus (58) Google Scholar]. Symptoms arise in the first decade of life in 37%, but over half become symptomatic after 20 years of age.Although chest radiographs can identify findings suspicious of bronchopulmonary sequestration in most cases, CT angiography is the preferred imaging modality for identifying the anomaly and its aberrant arteries [7Abbey P. Das C.J. Pangtey G.S. Seith A. Dutta R. Kumar A. Imaging in bronchopulmonary sequestration.J Med Imaging Radiat Oncol. 2009; 53: 22-31Crossref PubMed Scopus (58) Google Scholar]. Magnetic resonance angiography may be used where CT is not desirable and invasive catheter angiography could be considered if previous methods fail to identify the vascular supply of the sequestration.Surgical resection is recommended for bronchopulmonary sequestration to avoid future infection and hemorrhage [1Savic B. Birtel F.J. Tholen W. Funke H.D. Knoche R. Lung sequestration: report of seven cases and review of 540 published cases.Thorax. 1979; 34: 96-101Crossref PubMed Scopus (517) Google Scholar, 7Abbey P. Das C.J. Pangtey G.S. Seith A. Dutta R. Kumar A. Imaging in bronchopulmonary sequestration.J Med Imaging Radiat Oncol. 2009; 53: 22-31Crossref PubMed Scopus (58) Google Scholar]. The numbers and position of aberrant arteries supplying the resection should be carefully noted as inadvertent injury could cause massive hemorrhage. Intralobar sequestrations usually require segmentectomy or lobectomy. Video-assisted thoracic surgery offers an alternative less invasive approach to conventional thoracotomy for sequestration resection [3de Lagausie P. Bonnard A. Berrebi D. Petit P. Dorgeret S. Guys J.M. Video-assisted thoracoscopic surgery for pulmonary sequestration in children.Ann Thorac Surg. 2005; 80: 1266-1269Abstract Full Text Full Text PDF PubMed Scopus (39) Google Scholar, 4Tsang F.H. Chung S.S. Sihoe A.D. Video-assisted thoracic surgery for bronchopulmonary sequestration.Interact Cardiovasc Thorac Surg. 2006; 5: 424-426Crossref PubMed Scopus (28) Google Scholar, 5Kestenholz P.B. Schneiter D. Hillinger S. Lardinois D. Weder W. Thoracoscopic treatment of pulmonary sequestration.Eur J Cardiothorac Surg. 2006; 29: 815-818Crossref PubMed Scopus (79) Google Scholar, 7Abbey P. Das C.J. Pangtey G.S. Seith A. Dutta R. Kumar A. Imaging in bronchopulmonary sequestration.J Med Imaging Radiat Oncol. 2009; 53: 22-31Crossref PubMed Scopus (58) Google Scholar]. However, factors like dense adhesions clearly could make VATS more difficult to perform, and in these cases a thoracotomy may be required. Preoperative arterial embolization of the sequestration's aberrant blood supply has been described and may mitigate the risk of dissection and division of the systemic arterial supply [8Saxena P. Marshall M. Ng L. Sinha A. Edwards M. Preoperative embolization of aberrant systemic artery in sequestration of lung.Asian Cardiovasc Thorac Ann. 2011; 19: 357-359Crossref PubMed Scopus (10) Google Scholar].The coexistence of lung malignancy and bronchopulmonary sequestration is extremely rare, with only 9 previous cases reported in the literature; all of which were intralobar, 6 within the same lobe as the sequestration, and 3 were adenocarcinoma [2Lawal L. Mikroulis D. Eleftheriadis S. Karros P. Bougioukas I. Bougioukas G. Adenocarcinoma in pulmonary sequestration.Asian Cardiovasc Thorac Ann. 2011; 19: 433-435Crossref PubMed Scopus (15) Google Scholar]. We present the first case of VATS lobectomy of sequestration and cancer. Where lung malignancy is suspected in patients with bronchopulmonary sequestration, biopsy and staging should be performed to clarify the diagnosis and subsequent management. Due to the rarity of malignancy coexisting with sequestration, however, full malignancy workup is not routinely recommended for all bronchopulmonary sequestration [2Lawal L. Mikroulis D. Eleftheriadis S. Karros P. Bougioukas I. Bougioukas G. Adenocarcinoma in pulmonary sequestration.Asian Cardiovasc Thorac Ann. 2011; 19: 433-435Crossref PubMed Scopus (15) Google Scholar].Bronchopulmonary sequestration is very uncommon and is extremely rare to coexist with lung cancer. This is the first report of thoracoscopic resection of a synchronous lung cancer and pulmonary sequestration. Importantly, the case demonstrates the use of an endoscopic stapler to divide the aberrant arterial supply to the sequestration and facilitate minimally invasive resection. Further, this report demonstrates the coexistence of lung malignancy and sequestration should not exclude a minimally invasive approach. Bronchopulmonary sequestration makes up 0.15% to 6.4% of congenital pulmonary anomalies and is defined as an area of lung parenchyma not connected with the tracheobronchial system with its own aberrant systemic arterial supply, usually from the aorta [1Savic B. Birtel F.J. Tholen W. Funke H.D. Knoche R. Lung sequestration: report of seven cases and review of 540 published cases.Thorax. 1979; 34: 96-101Crossref PubMed Scopus (517) Google Scholar]. The coexistence of lung malignancy and bronchopulmonary sequestration is extremely rare [2Lawal L. Mikroulis D. Eleftheriadis S. Karros P. Bougioukas I. Bougioukas G. Adenocarcinoma in pulmonary sequestration.Asian Cardiovasc Thorac Ann. 2011; 19: 433-435Crossref PubMed Scopus (15) Google Scholar]. Thoracoscopic lobectomy for lung cancer is well established and thoracoscopic resection of bronchopulmonary sequestration has been previously reported [3de Lagausie P. Bonnard A. Berrebi D. Petit P. Dorgeret S. Guys J.M. Video-assisted thoracoscopic surgery for pulmonary sequestration in children.Ann Thorac Surg. 2005; 80: 1266-1269Abstract Full Text Full Text PDF PubMed Scopus (39) Google Scholar, 4Tsang F.H. Chung S.S. Sihoe A.D. Video-assisted thoracic surgery for bronchopulmonary sequestration.Interact Cardiovasc Thorac Surg. 2006; 5: 424-426Crossref PubMed Scopus (28) Google Scholar, 5Kestenholz P.B. Schneiter D. Hillinger S. Lardinois D. Weder W. Thoracoscopic treatment of pulmonary sequestration.Eur J Cardiothorac Surg. 2006; 29: 815-818Crossref PubMed Scopus (79) Google Scholar]. A 65-year-old man was incidentally found (on magnetic resonance imaging) to have a 5.5 × 3.5 cm cystic mass in the left hemithorax. Subsequent chest radiograph showed a left retrocardiac opacity and a computed tomography (CT) showed the presence of either a neuroenteric cyst due to its paraspinal location, or a bronchopulmonary sequestration. Follow-up CT demonstrated a new 2.5-cm spiculated nodule in the left lower lobe just medial to the cystic lesion and an arterial connection to the cystic lesion from the descending thoracic aorta just superior to the diaphragm, which provided convincing evidence of intralobar pulmonary sequestration (Fig 1). A CT-guided biopsy of the lung nodule established a histologic diagnosis of adenocarcinoma. Positron emission tomography confirmed stage 1A disease (T1BN0M0). Preoperative pulmonary function tests were within normal limits and the decision was made to proceed to resection of both lesions by video-assisted thoracoscopic surgery (VATS) left lower lobectomy. Three ports and a utility incision were made. After confirmation of the site of the sequestration and lung primary, the left lower lobe was retracted cephalad. The inferior pulmonary ligament was identified and the aberrant arterial supply to the sequestration noted (Fig 2). Careful dissection of the aberrant vessel was performed and a silk tie passed around it. The vessel was divided with an Endo-GIA vascular stapler (Covidien, Norwalk, CT). A Chitwood-DeBakey straight aortic cross clamp was positioned proximally on the aberrant vessel in the event of stapler misadventure. A “fissure-less” technique was used to complete the left lower lobectomy with sequential division using endoscopic staplers of the inferior pulmonary vein, the lower lobe bronchus, and the continuation of the pulmonary artery to the lower lobe. After the fissure was completed, the lobe was placed in a bag and removed through the utility incision. Operative time was 105 minutes. Blood loss was less than 50 mL and the patient made a routine uncomplicated recovery; he was discharged home on postoperative day 2. Figure 3 shows the resected lung specimen with the tumor and sequestration. CommentBronchopulmonary sequestration is classified anatomically as intralobar (∼75%) or extralobar (∼25%), depending on whether the sequestrated tissue shares the same pleural investment with normal lung tissue [1Savic B. Birtel F.J. Tholen W. Funke H.D. Knoche R. Lung sequestration: report of seven cases and review of 540 published cases.Thorax. 1979; 34: 96-101Crossref PubMed Scopus (517) Google Scholar]. It has a systemic aberrant arterial supply from the thoracic aorta 74% of the time, more than 1 aberrant artery in 15%, while venous return is to the pulmonary veins in 96%. Cystic changes, fibrosis and chronic inflammation of lung parenchyma are the main histologic features [6Stocker J.T. Sequestrations of the lung.Semin Diagn Pathol. 1986; 2: 106-121Google Scholar]. Clinical presentation can range from cough, hemoptysis, pneumonia, and congestive heart failure to potential associations with other pulmonary and skeletal anomalies such as congenital diaphragmatic hernia and scoliosis [1Savic B. Birtel F.J. Tholen W. Funke H.D. Knoche R. Lung sequestration: report of seven cases and review of 540 published cases.Thorax. 1979; 34: 96-101Crossref PubMed Scopus (517) Google Scholar, 7Abbey P. Das C.J. Pangtey G.S. Seith A. Dutta R. Kumar A. Imaging in bronchopulmonary sequestration.J Med Imaging Radiat Oncol. 2009; 53: 22-31Crossref PubMed Scopus (58) Google Scholar]. Symptoms arise in the first decade of life in 37%, but over half become symptomatic after 20 years of age.Although chest radiographs can identify findings suspicious of bronchopulmonary sequestration in most cases, CT angiography is the preferred imaging modality for identifying the anomaly and its aberrant arteries [7Abbey P. Das C.J. Pangtey G.S. Seith A. Dutta R. Kumar A. Imaging in bronchopulmonary sequestration.J Med Imaging Radiat Oncol. 2009; 53: 22-31Crossref PubMed Scopus (58) Google Scholar]. Magnetic resonance angiography may be used where CT is not desirable and invasive catheter angiography could be considered if previous methods fail to identify the vascular supply of the sequestration.Surgical resection is recommended for bronchopulmonary sequestration to avoid future infection and hemorrhage [1Savic B. Birtel F.J. Tholen W. Funke H.D. Knoche R. Lung sequestration: report of seven cases and review of 540 published cases.Thorax. 1979; 34: 96-101Crossref PubMed Scopus (517) Google Scholar, 7Abbey P. Das C.J. Pangtey G.S. Seith A. Dutta R. Kumar A. Imaging in bronchopulmonary sequestration.J Med Imaging Radiat Oncol. 2009; 53: 22-31Crossref PubMed Scopus (58) Google Scholar]. The numbers and position of aberrant arteries supplying the resection should be carefully noted as inadvertent injury could cause massive hemorrhage. Intralobar sequestrations usually require segmentectomy or lobectomy. Video-assisted thoracic surgery offers an alternative less invasive approach to conventional thoracotomy for sequestration resection [3de Lagausie P. Bonnard A. Berrebi D. Petit P. Dorgeret S. Guys J.M. Video-assisted thoracoscopic surgery for pulmonary sequestration in children.Ann Thorac Surg. 2005; 80: 1266-1269Abstract Full Text Full Text PDF PubMed Scopus (39) Google Scholar, 4Tsang F.H. Chung S.S. Sihoe A.D. Video-assisted thoracic surgery for bronchopulmonary sequestration.Interact Cardiovasc Thorac Surg. 2006; 5: 424-426Crossref PubMed Scopus (28) Google Scholar, 5Kestenholz P.B. Schneiter D. Hillinger S. Lardinois D. Weder W. Thoracoscopic treatment of pulmonary sequestration.Eur J Cardiothorac Surg. 2006; 29: 815-818Crossref PubMed Scopus (79) Google Scholar, 7Abbey P. Das C.J. Pangtey G.S. Seith A. Dutta R. Kumar A. Imaging in bronchopulmonary sequestration.J Med Imaging Radiat Oncol. 2009; 53: 22-31Crossref PubMed Scopus (58) Google Scholar]. However, factors like dense adhesions clearly could make VATS more difficult to perform, and in these cases a thoracotomy may be required. Preoperative arterial embolization of the sequestration's aberrant blood supply has been described and may mitigate the risk of dissection and division of the systemic arterial supply [8Saxena P. Marshall M. Ng L. Sinha A. Edwards M. Preoperative embolization of aberrant systemic artery in sequestration of lung.Asian Cardiovasc Thorac Ann. 2011; 19: 357-359Crossref PubMed Scopus (10) Google Scholar].The coexistence of lung malignancy and bronchopulmonary sequestration is extremely rare, with only 9 previous cases reported in the literature; all of which were intralobar, 6 within the same lobe as the sequestration, and 3 were adenocarcinoma [2Lawal L. Mikroulis D. Eleftheriadis S. Karros P. Bougioukas I. Bougioukas G. Adenocarcinoma in pulmonary sequestration.Asian Cardiovasc Thorac Ann. 2011; 19: 433-435Crossref PubMed Scopus (15) Google Scholar]. We present the first case of VATS lobectomy of sequestration and cancer. Where lung malignancy is suspected in patients with bronchopulmonary sequestration, biopsy and staging should be performed to clarify the diagnosis and subsequent management. Due to the rarity of malignancy coexisting with sequestration, however, full malignancy workup is not routinely recommended for all bronchopulmonary sequestration [2Lawal L. Mikroulis D. Eleftheriadis S. Karros P. Bougioukas I. Bougioukas G. Adenocarcinoma in pulmonary sequestration.Asian Cardiovasc Thorac Ann. 2011; 19: 433-435Crossref PubMed Scopus (15) Google Scholar].Bronchopulmonary sequestration is very uncommon and is extremely rare to coexist with lung cancer. This is the first report of thoracoscopic resection of a synchronous lung cancer and pulmonary sequestration. Importantly, the case demonstrates the use of an endoscopic stapler to divide the aberrant arterial supply to the sequestration and facilitate minimally invasive resection. Further, this report demonstrates the coexistence of lung malignancy and sequestration should not exclude a minimally invasive approach. Bronchopulmonary sequestration is classified anatomically as intralobar (∼75%) or extralobar (∼25%), depending on whether the sequestrated tissue shares the same pleural investment with normal lung tissue [1Savic B. Birtel F.J. Tholen W. Funke H.D. Knoche R. Lung sequestration: report of seven cases and review of 540 published cases.Thorax. 1979; 34: 96-101Crossref PubMed Scopus (517) Google Scholar]. It has a systemic aberrant arterial supply from the thoracic aorta 74% of the time, more than 1 aberrant artery in 15%, while venous return is to the pulmonary veins in 96%. Cystic changes, fibrosis and chronic inflammation of lung parenchyma are the main histologic features [6Stocker J.T. Sequestrations of the lung.Semin Diagn Pathol. 1986; 2: 106-121Google Scholar]. Clinical presentation can range from cough, hemoptysis, pneumonia, and congestive heart failure to potential associations with other pulmonary and skeletal anomalies such as congenital diaphragmatic hernia and scoliosis [1Savic B. Birtel F.J. Tholen W. Funke H.D. Knoche R. Lung sequestration: report of seven cases and review of 540 published cases.Thorax. 1979; 34: 96-101Crossref PubMed Scopus (517) Google Scholar, 7Abbey P. Das C.J. Pangtey G.S. Seith A. Dutta R. Kumar A. Imaging in bronchopulmonary sequestration.J Med Imaging Radiat Oncol. 2009; 53: 22-31Crossref PubMed Scopus (58) Google Scholar]. Symptoms arise in the first decade of life in 37%, but over half become symptomatic after 20 years of age. Although chest radiographs can identify findings suspicious of bronchopulmonary sequestration in most cases, CT angiography is the preferred imaging modality for identifying the anomaly and its aberrant arteries [7Abbey P. Das C.J. Pangtey G.S. Seith A. Dutta R. Kumar A. Imaging in bronchopulmonary sequestration.J Med Imaging Radiat Oncol. 2009; 53: 22-31Crossref PubMed Scopus (58) Google Scholar]. Magnetic resonance angiography may be used where CT is not desirable and invasive catheter angiography could be considered if previous methods fail to identify the vascular supply of the sequestration. Surgical resection is recommended for bronchopulmonary sequestration to avoid future infection and hemorrhage [1Savic B. Birtel F.J. Tholen W. Funke H.D. Knoche R. Lung sequestration: report of seven cases and review of 540 published cases.Thorax. 1979; 34: 96-101Crossref PubMed Scopus (517) Google Scholar, 7Abbey P. Das C.J. Pangtey G.S. Seith A. Dutta R. Kumar A. Imaging in bronchopulmonary sequestration.J Med Imaging Radiat Oncol. 2009; 53: 22-31Crossref PubMed Scopus (58) Google Scholar]. The numbers and position of aberrant arteries supplying the resection should be carefully noted as inadvertent injury could cause massive hemorrhage. Intralobar sequestrations usually require segmentectomy or lobectomy. Video-assisted thoracic surgery offers an alternative less invasive approach to conventional thoracotomy for sequestration resection [3de Lagausie P. Bonnard A. Berrebi D. Petit P. Dorgeret S. Guys J.M. Video-assisted thoracoscopic surgery for pulmonary sequestration in children.Ann Thorac Surg. 2005; 80: 1266-1269Abstract Full Text Full Text PDF PubMed Scopus (39) Google Scholar, 4Tsang F.H. Chung S.S. Sihoe A.D. Video-assisted thoracic surgery for bronchopulmonary sequestration.Interact Cardiovasc Thorac Surg. 2006; 5: 424-426Crossref PubMed Scopus (28) Google Scholar, 5Kestenholz P.B. Schneiter D. Hillinger S. Lardinois D. Weder W. Thoracoscopic treatment of pulmonary sequestration.Eur J Cardiothorac Surg. 2006; 29: 815-818Crossref PubMed Scopus (79) Google Scholar, 7Abbey P. Das C.J. Pangtey G.S. Seith A. Dutta R. Kumar A. Imaging in bronchopulmonary sequestration.J Med Imaging Radiat Oncol. 2009; 53: 22-31Crossref PubMed Scopus (58) Google Scholar]. However, factors like dense adhesions clearly could make VATS more difficult to perform, and in these cases a thoracotomy may be required. Preoperative arterial embolization of the sequestration's aberrant blood supply has been described and may mitigate the risk of dissection and division of the systemic arterial supply [8Saxena P. Marshall M. Ng L. Sinha A. Edwards M. Preoperative embolization of aberrant systemic artery in sequestration of lung.Asian Cardiovasc Thorac Ann. 2011; 19: 357-359Crossref PubMed Scopus (10) Google Scholar]. The coexistence of lung malignancy and bronchopulmonary sequestration is extremely rare, with only 9 previous cases reported in the literature; all of which were intralobar, 6 within the same lobe as the sequestration, and 3 were adenocarcinoma [2Lawal L. Mikroulis D. Eleftheriadis S. Karros P. Bougioukas I. Bougioukas G. Adenocarcinoma in pulmonary sequestration.Asian Cardiovasc Thorac Ann. 2011; 19: 433-435Crossref PubMed Scopus (15) Google Scholar]. We present the first case of VATS lobectomy of sequestration and cancer. Where lung malignancy is suspected in patients with bronchopulmonary sequestration, biopsy and staging should be performed to clarify the diagnosis and subsequent management. Due to the rarity of malignancy coexisting with sequestration, however, full malignancy workup is not routinely recommended for all bronchopulmonary sequestration [2Lawal L. Mikroulis D. Eleftheriadis S. Karros P. Bougioukas I. Bougioukas G. Adenocarcinoma in pulmonary sequestration.Asian Cardiovasc Thorac Ann. 2011; 19: 433-435Crossref PubMed Scopus (15) Google Scholar]. Bronchopulmonary sequestration is very uncommon and is extremely rare to coexist with lung cancer. This is the first report of thoracoscopic resection of a synchronous lung cancer and pulmonary sequestration. Importantly, the case demonstrates the use of an endoscopic stapler to divide the aberrant arterial supply to the sequestration and facilitate minimally invasive resection. Further, this report demonstrates the coexistence of lung malignancy and sequestration should not exclude a minimally invasive approach.
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