Video-assisted thoracoscopic lobectomy in the treatment of intralobar pulmonary sequestration

Pulmonary sequestration is a congenital malformation characterised by cystic, non-functioning embryonic lung tissue with vascularisation of an abnormal systemic artery. They are classified as intralobar (75%) and extralobar (25%) and are more common in the left lung and lower lobes (60-90%). We report two cases of intralobar pulmonary sequestration located in the lower lobe of the left lung which were subjected to video-assisted thoracoscopic surgery (VATS). Both patients had recurrent infections for which, after performing imaging tests, they were diagnosed with intralobar pulmonary sequestration in the left lower lobe, with an afferent arterial branch to the malformation from the aorta. A lower lobectomy was performed by video-assisted surgery, dividing the aberrant aortic artery with an endostapler. A single thoracic chest tube was placed and removed on postoperative day 2 and the patients were discharged on the same day. In both cases, the pathology examination revealed intralobar pulmonary sequestration. Pulmonary sequestrations are uncommon malformations that can be operated on using minimally invasive techniques, thereby permitting early discharge and a low rate of complications.     

Techniques for Treating Aberrant Arteries During Resection of Pulmonary Sequestration by Video-Assisted Thoracic Surgery: Report of Two Cases

We describe the techniques we used for treating aberrant arteries during resection of pulmonary sequestration by video-assisted thoracic surgery (VATS) in two patients. In patient 1, the aberrant artery was transected after securing six rows of staples with a knifeless vascular endostapler. In patient 2, the aberrant artery was cut after ligation with special forceps that designed by one of us (S.K.) to push a knot, tied outside the body, into the thoracic cavity, then ligate the suture. Left lower lobectomy and right basal segmentectomy were both successfully performed by these methods. These two cases are reported to show that VATS lobectomy is a feasible and minimally invasive technique of treating pulmonary sequestration and other diseases of the lung. Received: October 12, 2001 / Accepted: July 2, 2002 Reprint requests to: S. Kaseda     

Pulmonary sequestration associated with asymptomatic aspergillosis.

Intralobar pulmonary sequestration associated with asymptomatic aspergillosis is a rare case. We describe the case of a 65-year-old woman with intrapulmonary sequestration, anomalous systemic arterial supply to the left lower lobe and aspergillosis who underwent left lower lobectomy and ligation of an anomalous artery by Video-Assisted Thoracoscopic surgery (VATS). Pathological examination showed the parenchymal distortion and chronic inflammation. Aspergillus were found in the cyst. VATS lobectomy for intralobar pulmonary sequestration is a safe and valid procedure.     

Extralobar pulmonary sequestration with aberrant lobulation of the lower lobe; report of a case

An adult case of extralobar pulmonary sequestration with aberrant lobulation of the right lower lobe is reported. A 32-year-old woman admitted to our hospital with a right intrathoracic tumor in the chest computed tomography (CT). Chest CT showed a mass lesion on the right diaphragm of a diameter 5 cm and abnormal fissure of the right lower lobe. Under the diagnosis of pulmonary sequestration without clearly thick vessels, video-assisted thoracoscopic surgery was performed. The mass existed between the right diaphragm and posterior lower lobe with a thin stalk, and the lower lobe had aberrant lobulation. The resected mass was histopathologically diagnosed as extralobar pulmonary sequestration.     

Video-assisted thoracic resection for intralobar pulmonary sequestration

We present a case in which video-assisted thoracic resection for intralobar pulmonary sequestration (ILPS) was successfully performed. A 36-year-old woman had repeated pneumonia. Chest computed tomography (CT) showed a round mass in the right lower lobe of the lung. Subsequent three-dimensional CT revealed that a large anomalous artery arising from the descending thoracic aorta was distributing to the posterior basal segment containing the lesion and was draining into the inferior pulmonary vein. The patient was diagnosed with ILPS and underwent surgery. The anomalous artery was divided, and the sequestered segment was completely resected by video-assisted thoracic surgery (VATS). We think that VATS resection for ILPS is feasible and is a major therapeutic option as noninvasive surgery.     

A case report of pulmonary sequestration (Pryce I) associated with infective endocarditis

We experienced a case of pulmonary sequestration of Pryce type I associated with infective endocarditis (IE). A 19-old-man had prolonged high fever of 39 degrees C against antibiotic therapy. He was referred to our hospital because of the positive blood culture and abnormal echocardiographic findings, which were severe aortic regurgitation with vegetations clinging the aortic cusps. In addition, his chest X-ray film showed mass lesion behind the cardiac shadow, and continuous murmur was auscultated on this portion. The left pulmonary arteriography revealed no arterial distribution to the left lower lobe, while aortography showed an aberrant artery arising from the descending aorta entering into this lobe. One month after aortic valve replacement for IE, left lower lobectomy and amputation of the aberrant artery were performed successfully. Pathologically, inflammatory changes of the aortic valve and proliferations of intimal and medial wall of the aberrant artery were shown. However, alveo-bronchial structure of the resected lobe was normal. Diagnosis, complications and surgical management of pulmonary sequestration were discussed.     

Congenital cystic adenomatoid malformation connected to an extralobar pulmonary sequestration in the contralateral chest: common origin?

Extralobar pulmonary sequestration (ELS) and congenital cystic adenomatoid malformation have been reported to coexist in several variations. This suggests a common embryologic origin. A 6-month-old boy presented with a history of recurrent pneumonias. The patient was diagnosed with a right lower lobe congenital cystic adenomatoid malformation (CCAM) and a left lower lobe ELS/CCAM. The diagnosis was made with the aid of a multidetector computed tomography (CT). Three-dimensional CT reconstruction showed the presence of a right lower lobe CCAM, a left lower ELS with an aberrant arterial supply from the celiac axis, and possible venous drainage into the right CCAM. The patient underwent a right thoracotomy. Intraoperatively, the lesions were discovered to be connected by a band of tissue. The right lower lobe CCAM and the left ELS were removed from the right chest. Histologic analysis confirmed the presence of a CCAM within the right lower lobe. The ELS had involvement of a type II CCAM within the sequestration. The connection between the right CCAM and left ELS/CCAM showed an anomalous conducting airway, anomalous vein, and anomalous artery connecting the 2 lesions. The authors present the first case of a CCAM connected to an ELS/CCAM in the contralateral hemithorax. The unique anatomic configuration of these lesions suggests a common embryologic origin of ELS and CCAM.     

Thoracoscopic treatment of pulmonary sequestration.

OBJECTIVE: Pulmonary sequestration is a rare congenital malformation and may be the cause of recurrent infections or hemoptysis. It has been shown in case reports that resection by video-assisted thoracic surgery (VATS) is feasible despite the possible technical difficulties due to inflammatory changes, but its role has not been evaluated yet in a larger series of consecutively treated patients. METHODS: Retrospective analysis of all consecutively thoracoscopically treated patients (between January 1991 and January 2005) with pulmonary sequestration in a single center. We included 14 patients in the study who fulfilled the criteria; seven were women. Median age was 33 years (20-64 years). The following data were analyzed for all patients: major symptoms, diagnostic procedures, treatment, and outcome. Operative parameters and findings including operating time, blood loss, anatomical location of the sequestration, and feeding vessels were evaluated. RESULTS: Leading symptoms were recurrent infections (10), hemoptysis (3), and chest discomfort (1). The diagnosis was made by CT scan. Additionally, an arteriography or an angio-MRI was done in three patients and one patient, respectively. Thirteen intralobar (all lower lobes, eight on the right) and one left-sided extralobar pulmonary sequestration were resected. We performed eight lobectomies, four atypical segmentectomies, one extralobar resection, and one occlusion of the aberrant artery. One case had to be converted to a thoracotomy due to bleeding from the aberrant artery. There was no mortality. Complications included pneumonia in three cases, one hemothorax, one pneumothorax after removing the chest tube, and one wound infection. All were treated conservatively. CONCLUSION: Thoracoscopic treatment of pulmonary sequestration is feasible in experienced hands. The aberrant systemic artery can be freed and dissected safely despite the frequently occurring inflammatory changes. Conversion rate to thoracotomy is low.     

Systemic origin of the sole artery to the basal segments of the left lung without pulmonary sequestration

A sixty-one year-old man with squamous cell carcinoma of the left upper lobe had an aberrant systemic artery to the left basal segments without pulmonary sequestration. Physical examination revealed neither cardiac murmur nor any sign of heart failure, which was at variance with reported cases in the literature. Chest X-ray film showed no abnormal density suggesting sequestrated lung. Bronchogram disclosed obstruction of the left upper lobar bronchus by the tumor and normal segmental bronchi of the lower lobe. Left pulmonary angiogram showed normal arterial distribution of the left upper lobe and the superior segment of the lower lobe, but the basal segmental arteries were not visualized. The aberrant pulmonary artery arising from the descending aorta was visualized by computed tomography. Following dissection of the abnormal vessel pneumonectomy was performed. Pathological examination of the left basal segments revealed prominent atheromatous changes in the aberrant systemic intrapulmonary artery and irreversible obstructive lesions in its tributaries. These arterial lesions in this patient would have precluded plastic operations such as transfer of the origin of the aberrant vessel to the left pulmonary artery even if other circumstances had been favorable for preservation of the left lower lobe.     

Coil embolization to treat pulmonary sequestration in the right upper lobe

Although there have been a few case reports of pulmonary sequestration, it is primarily located in the lower lobe and left lung, rarely in the right upper lobe. Here, we report a case presented with haemoptysis. Computed tomography images revealed flake ground-glass shadows in the right upper lobe. Computed tomography angiography demonstrated an artery supplied the affected lesions stemmed from the aortic arch. We diagnosed and treated her with bronchial artery angiography with coil embolization. No complications were found after operation until now. Thus, CTA could help identify the abnormal blood vessels, and interventional therapy may be an effective alternative to surgery of pulmonary sequestration.     

An incidental and uncommon pulmonary sequestration with an uncommon feeding artery

INTRODUCTIONPulmonary sequestration is a rare congenital anomaly and most intralobar sequestrations were located in lower lobes. There is little information on middle lobe intralobar sequestration.PRESENTATION OF CASEA 44 year-old man with right middle lobe syndrome was referred for surgical management. He underwent video-assisted thoracoscopic right middle lobectomy and his postoperative course was uneventful. Pathology is notable for an incidental intralobar sequestration.DISCUSSIONOur case was unique in that the location of incidental pulmonary sequestration is uncommon and in that its feeding artery was uncommonly located in the fissure.CONCLUSIONWe report an extremely rare case of right middle lobe intralobar sequestration with a feeding artery in the fissure.     

Neonatal hyperreninemia and hypertensive heart failure relieved with resection of an intralobar pulmonary sequestration

Intralobar pulmonary sequestrations are congenital lung malformations sometimes known to cause symptoms of heart failure in the neonate because of shunting of blood flow from an anomalous systemic feeding artery into the pulmonary venous system. These are cured by pulmonary lobectomy. We report the case of a 37-weeks' gestational age infant who presented with bilateral pleural effusions perinatally, followed by hypertensive heart failure and hyperreninemia 2 weeks postnatally. Computed tomography revealed a large artery from the infradiaphragmatic aorta proximal to the celiac axis supplying a right lower lobe intralobar pulmonary sequestration and 4 small nonstenosed renal arteries. Subsequent to a right lower lobectomy, the patient recovered with a precipitous drop in plasma renin assay level and was discharged from the neonatal intensive care unit in good condition 12 days postoperatively. This is the first reported case of hyperreninemia treated by lobectomy. We hypothesize that the aberrant feeding vessel resulted in a relative deficiency in renal perfusion via a “steal” effect that was relieved by surgical ligation of the artery.     

Surgical treatment of infected intralobar pulmonary sequestration: a collective review of patients older than 50 years reported in the literature.

We report on the rare and surgical treatment of a senile patient of infected intralobar pulmonary sequestration. A 56-year-old male who had complained of headache, vomiting, cough, sputum production, and high fever was admitted to our hospital. Chest computed tomography (CT) showed an infected intralobar pulmonary sequestration as an 8x6 cm cystic mass with multiple air-fluid cavities in the left lower basal segment and severe pneumonia in the left upper and lower lobes around the mass. A 3-D CT showed an aberrant artery entering the consolidation from the descending aorta. A standard lower lobectomy was performed with a ligation of the aberrant artery with a diameter of 1 cm supplying the posterior segment of the left lower lobe. A histological examination of the lung revealed acute and chronic broncho-bronchiolitis with cystic dilatation consistent with intralobar pulmonary sequestration. We discuss the characters of senile patients compared with juvenile patients, with reference to a collective review of patients older than 50 reported in the literature.     

Anomalous systemic arterial supply to normal basal segments of the left lower lobe.

BACKGROUND: Anomalous arterial supply to the normal basal segments of the lower lobe without sequestration is a rare congenital abnormality, and whether it belongs to the broad spectrum of sequestration disorders remains controversial. METHODS: The cases of all 4 patients who were treated surgically by us were reviewed together with 8 previously reported cases. RESULTS: The anomalous artery originated from the descending thoracic aorta, distributed to the basal segments of the left lower lobe, and drained to a normal inferior pulmonary vein in each case. The anomalous artery was thick and elastic walled. From the review of all 12 cases, male gender, left side, descending thoracic aorta as the aberrant arterial origin, absence of pulmonary blood flow to the basal segments, and normal pulmonary venous drainage were predominant. Despite some differences, the findings seemed closely related to intralobar sequestration. Surgical treatments were lung resection, anastomosis, and ligation of the anomalous artery. CONCLUSIONS: This anomaly is probably one type of sequestration complex. Both aortic and pulmonary arterial angiographic studies are needed to plan the definitive surgical procedure.     

Upper thoracic extralobar pulmonary sequestration with anomalous blood supply from the subclavian artery

The authors report on a newborn boy with extralobar pulmonary sequestration in the right upper thoracic region. Preoperative angiography showed an anomalous large vessel from the right subclavian artery, supplying the sequestrated lobe. Right thoracotomy was performed to resect the sequestrated lobe, which was diagnosed as extralobar form. This is an extremely rare case of extralobar pulmonary sequestration in which anomalous blood supply from the subclavian artery was seen preoperatively on radiographs. The authors recommend angiographic examination, particularly in case of diagnostic difficulty.     

Pulmonary sequestration associated with aspergillosis.

Pulmonary sequestration involves an abnormal pulmonary tissue separated from the normal pulmonary parenchyma, not connected to the tracheobronchial tree and supplied by a systemic artery. A case of intralobar pulmonary sequestration is presented. Case; a 49-year-old male was admitted to our hospital complaining of fever, cough and sputum production. Sputum culture resulted in a large growth of Aspergillus niger. Angiography showed an abnormal blood supply from the abdominal aorta to the right lower lobe. Right lower lobectomy was performed. The postoperative courses are uneventful.     

Intrapulmonary sequestration with arterial supply from the left internal thoracic artery: a case report.

Pulmonary sequestration is uncommon in the upper lobe. Its arterial supply from the internal thoracic artery is very rare. Reported here is a case of a 20-year-old male whose presenting symptom was recurrent pneumonia. Helical computed tomography (CT) and three-dimensional reconstruction images showed that aberrant arteries arising from the left internal thoracic artery were supplying the area of sequestration and draining into the pulmonary vein. Selective intra-arterial digital substraction angiogram also showed left internal thoracic artery supplying the area of the sequestration. Helical three-dimensional CT is noninvasive and provides as accurate three-dimensional information of the aberrant vascular supply in intrapulmonary sequestration as the angiography.     

Unique extralobar sequestration with atypical location and aberrant vessels

We report a unique extralobar pulmonary sequestration in the upper thoracic region with 3 aberrant vessels connecting to the right subclavian artery, right superior pulmonary vein, and right pulmonary artery in a 20-year-old man. The sequestered lung was completely excised using a video-assisted thoracoscopic approach.     

Video-assisted thoracic surgery lobectomy for lung cancer in a patient with complete situs inversus

Complete situs inversus is a rare abnormality of autosomal recessive inheritance; it requires particular care during surgery, because the viscus anatomy is a mirror image of the normal anatomy. Reports of surgery for lung cancer in cases of complete situs inversus are very rare. Here, we report a case of lobectomy for lung cancer of the right lower lobe performed using video-assisted thoracic surgery (VATS) in a patient with complete situs inversus. We emphasize the importance of careful examination of the relationship between the bronchus, pulmonary artery, and pulmonary vein in the hilum of the lung in cases of complete situs inversus requiring lung resection for cancer; this is even more necessary when VATS is performed.     

A case of intralobar pulmonary sequestration associated with abnormal connection between the aberrant artery and the pulmonary artery

The patient was a 5-year-old girl and had a history of recurrent pneumonia. Angiogram demonstrated that the aberrant arteries arising from descending aorta communicated with left pulmonary A10 where the multi-lobulated cystic lesion was seen by chest X-P. The right heart catheterization revealed the step-up of oxygen saturation in distal part of left pulmonary artery A10. Diagnosis was the intralobar pulmonary sequestration with the aberrant arteries communicating with pulmonary artery. The connection between aberrant arteries and pulmonary artery in this case might be caused by the chronic inflammation in the sequestrated lung. We found 4 case reports in which the communication between aberrant arteries and pulmonary artery was evident.