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.     

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.     

Diagnosis and management of congenital cystic disease of the lung in children

Pulmonary sequestration, congenital adenomatoid malformation (CAM), congenital lobar overinflation (CLO), and bronchogenic cysts are four congenital lesions that may present as abnormal cystic areas within the pleural cavity in early life. They share similar clinical and embryologic characteristics, are frequently difficult to diagnose, and all require surgical treatment. From December 1974 to January 1985, 22 patients were operated upon on the Pediatric Surgical Service for congenital cystic disease of the lung. There were eight females and 14 males, ranging in age from 1 day to 18 years; 12 were under 6 months of age, and 14 were under one year. There were 7 CAMs, 5 extralobar pulmonary sequestrations, 5 intralobar pulmonary sequestrations, 3 bronchogenic cysts, and 3 cystic lobes caused by CLO. One patient had bilateral pulmonary sequestrations, intralobar on one side and extralobar on the other, with both sharing a common systemic artery arising from the infradiaphragmatic aorta. All except two asymptomatic patients presented with either progressive respiratory distress or recurrent pulmonary infections, and had cystic changes noted on chest roentgenograms. The differentiation of these congenital lesions from resolvable inflammatory cysts is important. Diagnosis was aided by selective utilization of barium contrast studies, ultrasonography, computed tomography, and arteriography. Operation consisted of resection alone of the extralobar pulmonary sequestrations and bronchogenic cysts, and total lobectomy in CAM, intralobar sequestration, and CLO. One infant with CAM died 1 day postoperatively from bilateral hypoplastic lungs. The other 21 patients are alive and well with follow-up ranging from 1 month to 9 years, with a mean of 3.5 years.(ABSTRACT TRUNCATED AT 250 WORDS)     

Late revelation of a subphrenic extralobar pulmonary sequestration as a suprarenal mass

Pulmonary sequestrations are some rare congenital anomalies. The incidence was estimated of 0.15% to 1.7%. They are characterized by a mass of non functioning pulmonary tissue that has no communication to the normal bronchial tree. The vascularisation is supplied by systemic arteries. They are classified further as intralobar and extralobar types. Extralobar sequestration, so-called accessory lung, is separated from the normal lung. We present a rare case of subphrenic extralobar pulmonary sequestration in a 57 years old patient. The lesion was initially presented as a non-typical suprarenal mass discovered on CT scan. The approach by laparatomy permitted the resection and the definitive diagnosis.     

Thoracoscopic resection of an apical extralobar pulmonary sequestration in an infant

Pulmonary sequestration is a form of bronchopulmonary-foregut malformation that is treated with surgical resection, either via a thoracotomy or the thoracoscopic approach. Apical extralobar pulmonary sequestrations are rare. We report a case of an apical pulmonary sequestration in an infant that was amenable to thoracoscopic resection. An 8-month-old girl, weighing 10.3 kg, with a left apical extralobar pulmonary sequestration underwent thoracoscopic resection. The pulmonary sequestration was identified and dissected free from surrounding tissue. The vascular peduncle was secured with an endo-GIA loaded with 2-mm vascular staples. The infant was discharged on postoperative day two without complications. In spite of the patient's small size and the apical nature of the sequestration, the operation was easily performed via a thoracoscopic approach. Thoracoscopic resection of an apical extrapulmonary sequestration in a small infant is feasible and may minimize the morbidity of this procedure.     

Preoperative diagnosis of extralobar pulmonary sequestration with unusual vasculature: a case report.

Pulmonary sequestrations are uncommon congenital malformations of the lung. Vascular supply to pulmonary sequestrations is variable, with many unusual combinations. Adequate preoperative evaluation is hence advisable to avoid intraoperative complications. Magnetic resonance imaging (MRI) allows accurate diagnosis of extralobar pulmonary sequestration (ELPS), including definition of vascular supply. This may obviate the need for more invasive investigations that were suggested in the past. Presented here is a case of ELPS with its vascular attachments to the intercostal vessels.     

The multiple facets of pulmonary sequestration

PURPOSE: The goal of this study was to identify the proportion of sequestrations that were atypical or associated with other entities, such as congenital cystic adenomatoid malformations, communicating bronchopulmonary foregut malformations, bronchogenic cyst, and scimitar syndrome. METHODS: All charts of patients with pulmonary sequestration admitted at 2 children's hospitals from 1982 to July 1999 were reviewed retrospectively. The authors included all anomalies with a systemic arterial supply or without bronchial connection. RESULTS: Only 22 of the 39 patients (56%) had a classic isolated extralobar or intralobar sequestration, whereas the others presented with a spectrum of anomalies. Of the 13 cases diagnosed prenatally, 85% were asymptomatic at birth. In contrast, 26 cases diagnosed postnatally were all symptomatic, with those patients less than 2 weeks old presenting with various degrees of respiratory distress, and those older than 2 weeks old presenting with respiratory infections. The correct diagnosis was made preoperatively in 59% of cases. Only 4 patients did not undergo resection of their lesion, of which, 1 underwent interventional radiology with embolization of the anomalous arterial supply. Follow-up issues of importance included pneumonia, asthma, gastroesophageal reflux, and pectus excavatum. CONCLUSIONS: Sequestrations represent a spectrum of anomalies that overlap with other lung lesions. To facilitate management, they should be described according to their (1) connection to the tracheobronchial tree, (2) visceral pleura, (3) arterial supply, (4) venous drainage, (5) foregut communication, (6) histology, (7) mixed/multiple lesions, and (8) whether there are associated anomalies. Surgeons should be aware that approximately 50% of sequestrations could be atypical or associated with other anomalies. This should be kept in mind when weighing the benefits of resection versus conservative management of pulmonary sequestrations.     

Intrapulmonary sequestration operated by video-assisted thoracoscopic surgery lobectomy; report of a case

Pulmonary sequestration is a rare malformation of the respiratory tract. We here report an adult case of intralobar pulmonary sequestration with aberrant artery of the right lower lobe. A 20-year-old man admitted to our hospital with a chest discomfort. Chest computed tomography (CT), angiography, and scintigraphy showed abnormal findings of the right lower lobe with an aberrant artery. Under the diagnosis of pulmonary sequestration, video-assisted thoracoscopic surgery (VATS) lobectomy was performed. VATS is useful for the operation of pulmonary sequestration.     

Intralobar Bronchopulmonary Sequestration with Mixed Venous Drainage in a 10-Year-Old Girl

Bronchopulmonary sequestration consists of a mass of abnormal lung tissue that has no normal connection with the bronchial tree and is supplied with blood from an aberrant artery mostly originating in the thoracic aorta. Two forms are recognized: intralobar and extralobar sequestration. The first is localized within the normal visceral pleura and has a venous drainage into the pulmonary system; the latter is localized without the normal lung in its own pleura with venous drainage into the systemic venous system. Intralobar sequestration is the most common form accounting for 75% of the cases. Intralobar sequestration usually presents in adolescence or adulthood with signs of recurrent pneumonia. Extralobar sequestration presents early in life with respiratory distress or feeding difficulties and is frequently associated with other congenital malformations. The diagnosis is confirmed by CT scan of the lungs and magnetic resonance angiography as demonstration of the aberrant vascular supply is essential for the diagnosis. Therapy consists in surgical removal.

We present a case of intralobar sequestration in a 10-year-old girl. The clinical symptomatology was typical. Arterial supply with two aberrant arteries and mixed venous drainage into the pulmonary and systemic systems were particular features.     

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.     

Bilateral intralobar pulmonary sequestration in a newborn, case report and review of the literature on bilateral pulmonary sequestrations

We describe a rare case of bilateral intralobar pulmonary sequestration in a newborn with both sequestrations vascularized from a vessel arising from the celiac trunk of the abdominal aorta. The larger sequestration on the right side was diagnosed antenatally, the left side postnatally. At the age of 7 months, the child underwent successful bilateral thoracotomy. In follow-up at the age of more than 1 year, no complications have occurred.     

Videothoracoscopic approach in the treatment of intralobar pulmonary sequestration. Report of a case

The Authors report on the use of a videothorascopic approach in the treatment of a patient with intralobar bronchopulmonary sequestration. The latter is a rare malformation which more often than not manifests itself in young patients with hemophthisis, recurrent infection and cough. The condition is difficult to diagnose and is often diagnosed intraoperatively. The treatment is surgical resection. The videothorascopic approach was used in a young patient presenting a lesion of the left inferior lobe with polycyclic contours suspected of being an intralobar bronchopulmonary sequestration, along with medical history findings of cough and hyperpyrexia. The approach permitted identification of the lesion in the context of the left inferior lobe, safe identification and section of the abnormal systemic vessel supplying the lesion and subsequent inferior lobectomy. The postoperative course was uneventful and the patient was discharged on postoperative day 5 with an excellent esthetic and functional outcome. The videothorascopic approach should be taken into consideration for the diagnosis and treatment of intralobar bronchopulmonary sequestrations.     

Lung sequestration: report of seven cases and review of 540 published cases

We present an analysis of 400 intralobar and 133 extralobar sequestrations, six cases presenting intra- and extralobar sequestration simultaneously, five sequestrations of one whole lung, and two bilateral intralobar sequestrations.     

Intralobar pulmonary sequestration with high level of serum CEA; report of a case

We reported a case of intralobar pulmonary sequestration with a high level of the serum CEA. A 53-year-old woman whose chief complaint was cough was admitted to our hospital. Enhanced chest computed tomography (CT) revealed the mass in the left lower lung, lymph-nodes swelling, and the aberrant artery. Magnetic resonance angiography (MRA) conformed the aberrant artery from the descending aorta. The level of serum CEA elevated at 9.6 ng/ml. Left lower lobectomy was performed. A diagnosis of intralobar pulmonary sequestration (Pryce type II) was established in this case. Histopathologically, the peribronchial epithelial cells in pulmonary sequestration showed weak positive for anti-CEA monoclonal antibody. Postoperative course was uneventful and the serum CEA level was 3.5 ng/ml in the normal range at the postoperative 17th day.     

Emergency surgical intervention after unsuccessful percutaneous transluminal angioplasty and stenting of aortic coarctation

Two adult patients were diagnosed with extralobar and intralobar pulmonary sequestration. One patient presented with haemoptysis. Both patients suffered from recurrent episodes of severe pulmonary infections. Both patients were treated by means of endovascular embolization using Amplatzer(?) vascular plugs (AVPs). They were discharged from hospital after 48 and 24 h and then followed up for 24 and six months, respectively. No recurrence of symptoms was observed. Computed tomography scans were obtained every six months. Persistent occlusion of vascular supply and moderate regression of the sequestered lung tissue are evident after 24 and six months in both patients. Just one case of an adult patient affected by pulmonary sequestration and treated by endovascular embolization has been reported to date. The present report is the first on the use of the AVPs in adults for this condition. The potential advantages and drawbacks of this treatment modality in adults are discussed, as well the specific benefit represented by the AVPs.     

Extralobar pulmonary sequestration with an unusual venous drainage to the portal vein: preoperative diagnosis and excision by video-assisted thoracoscopy

Pulmonary sequestration is an uncommon congenital malformation of the lung that can be classified as intralobar or extralobar (ELS). Approximately 90% of ELS occur in the left hemithorax. Approximately 10% of ELS may present below the diaphragm. Both types of sequestrations are characterized by pulmonary tissue that does not communicate with the bronchial tree. The arterial blood supply to 80% ELS is through a direct branch of the thoracic or abdominal aorta, in 15% via another systemic artery and 5% from the pulmonary artery. The venous drainage of ELS is variable, predominantly into the systemic circulation (via the azygos vein, hemiazygos vein, or inferior vena cava). Approximately 25% drain completely or partially through the pulmonary veins. We report an extremely rare case of ELS with unusual venous drainage to the portal vein.     

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.     

Intralobar pulmonary sequestration supplied by an anomalous aneurysmal artery

Pulmonary sequestration is a relatively rare condition in which a systemic artery supplies blood to an abnormal lung tissue. Pulmonary sequestration with an aneurysmal systemic artery is extremely rare. We describe the case of a 52-year-old man with intralobar pulmonary sequestration supplied by an aneurysmal systemic artery. Because the nomenclature of pulmonary sequestration is still not clear, we propose that type 1 intralobar pulmonary sequestration be called "systemic arterial supply to the normal lung," as named by many professionals, and for this to be distinguished from pulmonary sequestration.     

Combined endovascular and surgical approaches to treat intralobar pulmonary sequestration: a case report

Pulmonary sequestration is a congenital abnormality of a non-functional pulmonary mass with anomalous systemic arterial supply. Surgical resection is the gold standard treatment, but it carries a risk of life-threatening haemorrhage from accidental injury of the anomalous artery. Endovascular embolisation has been introduced as a safe alternative, but does not eliminate the possibility of symptom recurrence. We report a case of a 61-year old woman with intralobar pulmonary sequestration treated with a combination of endovascular coil embolisation and surgical resection.     

Resection of intralobar pulmonary sequestration after coil embolization of aberrant arteries: report of a case

Pulmonary sequestration describes a rare congenital mass of nonfunctional pulmonary tissue with an aberrant systemic arterial supply. Conventionally, the mass is removed surgically, but this carries a risk of serious and potentially fatal hemorrhage from the aberrant arteries during the operation. We describe how we performed coil embolization of the aberrant arteries immediately before surgery to overcome this problem. Coil embolization is safe, feasible and effective in preventing intraoperative bleeding. This case is reported to introduce a new preoperative procedure for pulmonary sequestration.