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.     

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.     

A case report of subdiaphragmatic pulmonary sequestration

A 32-year-old man with extralobar pulmonary sequestration under diaphragm is reported. The patient was admitted because of abnormal shadow on chest X-ray film without any complaints. Chest roentgenogram showed a homogenous density at the left cardiophrenic angle. Aortogram demonstrated an abnormal artery arising from the abdominal aorta to left subphrenic mass shadow. On laparotomy a large cystic mass connected with diaphragm was found behind the stomach and removed successfully. The postoperative course was uneventful. The histological diagnosis was subphrenic pulmonary sequestration. The frequency, localisation, pathogenesis of pulmonary sequestration are discussed.     

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 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.     

Extralobar pulmonary sequestration in a 4-months infant

Aim-Background

To highlight the rarity of a congenital condition of Pulmonary sequestration. This condition is presenting in two forms, intralobar and extralobar.

Case report

An extralobar sequestration in a 4-month-old infant is reported. A mass located in the left lower pulmonary lobe was diagnosed by a prenatal ultrasound examination at the sixth month of the pregnancy had set the possible diagnosis of cystic adenomatoid dysplasia of the left lower pulmonary lobe. No distress or other symptoms were observed. Surgical intervention with a left posterolateral thoracotomy over the fifth intercostal space allowed approach to the sequestration. The clinical presentation, differential diagnosis and therapeutic approach are discussed herein.

Conclusions

It is significant for the medical team to consider pulmonary sequestration in the differential diagnosis for any patient that presents with respiratory distress and a chest radiograph showing a mediastinal shift.     

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.     

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.     

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.     

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.     

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.     

Pulmonary sequestration.

Pulmonary sequestration is a congenital anomaly in which an aberrant systemic artery arising from the thoracic or upper abdominal aorta supplies part of the lungs, usually the lower lobe. The sequestered lung may be anatomically distinct from the remainder of the lobe (extralobar), or may be included in the substance of the lobe, in which case it may or may not have bronchial communication with the rest of the bronchial tree. The patients present, often in the first two decades of life, with recurrent and severe bronchopulmonary infections. Associated anomalies are present, especially in the extralobar variety. Nine cases of sequestration are reviewed, stressing significant clinical, radiological, and arteriographic findings. Preoperative demonstration of the anomalous vessel by aortography has contributed significantly to the planning and safety of the surgical procedure, which was generally a lower lobectomy. Eight out of nine patients survived the procedure.     

Malignant spindle cell tumour (fibrosarcoma) in sequestration of lung—a case report

Pulmonary sequestration is an uncommon congenital anomaly of respiratory system and usually diagnosed in childhood but can present at any at age. It frequently presents as a mediastinal mass with symptoms from vascular shunting, or associated anatomic defects. This case report describes an adult who presented with chest pain due to extralobar sequestration of right lung extending into the mediastinum. Per-operatively it was found to be an extra pulmonary mass lesion with a peduncle attached to right lung suggestive of extralobar sequestration. Histopathological examination of the specimen showed it to be spindle cell tumor (fibrosarcoma) in sequestrated lung. The patient had an uneventful recovery and is under regular follow up without any further problems.     

133. Arteriovenöses pulmonales Aneurysma im extralobär separierten Lungenlappen mit ungewöhnlich großem Links-Links-Shunt

Zusammenfassung Bei einem 13jährigen Knaben wurde ein mit einem L-LShunt von 40% des GKMV einhergehendes arteriovenöses, pulmonales Aneurysma, in einem separierten Lungenlappen des rechten Unterfeldes angetroffen, das von einem transdiaphragmal aus der Aorta abdominalis aufsteigenden Arterienast gespeist wurde. Gefäßanatomie und Hämodynamik wurden durch Farbstoff-Verdünnungsmethode, Cineangiokardiographie und transfemorale Aortographie abgeklärt. Die Therapie bestand in der Eastirpation des aneurysmatragenden, accemorischen Lungenanteils. Die Beobachtung ergänzt die bekannten Formen der Lungenseparation um einen vasculär-aneurysmatischen Typ.

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Arteriovenous pulmonary aneurysm localized in an extralobar sequestrated pulmonary lobe with an unusually large left-left shunt

Summary An arteriovenous pulmonary aneurysm in an eatralobar sequestrated pulmonary lobe of the lower right side causing a shunt volume of 40% of the systemic circulation volume was found in a 13-year-old boy in whom an atypically localized machinery murmur was detected. The aneurysm was fed by an arterial branch ascending from the abdominal aorta and passing across the diaphragm. Diagnosis was made by a dye-dilution method, cineangiocardiography and transfemoral aortography. The sequestrated lung lobe was resected. Our observation adds a vascular aneurysmatic type to the other known forms of pulmonary sequestration.

     

Intraabdominal pulmonary sequestration

Bronchopulmonary sequestration is an uncommon developmental abnormality that frequently presents as an incidental mass. Differential diagnosis includes malignancy. Reports of extralobar abdominal pulmonary sequestration in adults are sparse. Only 2.5% of all pulmonary sequestrations are detected below the diaphragm. The authors present a case of a 45-year-old woman and a literature review. Computed tomography, magnetic resonance imaging, and operative and pathologic findings are provided. A review of diagnosis and therapy is presented.     

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.     

Video-Assisted Thoracic Surgery Lobectomy for Extralobar Pulmonary Sequestration in a Child: Report of a Case

Pulmonary sequestration in infants and children is conventionally treated by resecting the sequestered lung parenchyma (sequestrectomy) or by performing lobectomy through a standard thoracotomy. We performed lobectomy by video-assisted thoracic surgery, using an original tracheal tube that we designed, in a 6-year-old boy with extralobar pulmonary sequestration and bronchiectasis in the left lower lobe.     

A unique congenital mediastinal malformation

We report a case of a 35-year-old patient presenting with a unique asymptomatic malformation associating extralobar pulmonary sequestration communicating with a bronchogenic cyst of the esophageal wall via the aortopulmonary window, dextroisomerism, and complete agenesia of the left pericardium. Despite computed tomography (CT) scan and magnetic resonance imaging (MRI), the diagnosis could not be established before left thoracotomy. The sequestrated lobe and bronchogenic cyst were then successfully resected.     

Extralobar pulmonary sequestration

Extralobar sequestration of pulmonary tissues is relatively rare and seldom produces symptoms. An incidental finding is described of extralobar sequestration of the lung in a Nigerian child, presenting as an anterior mediastinal polycystic mass. The mass was attached to the right pulmonary artery by a fibrovascular pedicle which contained a small elastic artery, veins, and nerve bundles. Sequestrated lobes are usually situated in the posterior basal lobes of the lungs and may be associated with other congenital abnormalities, but the case reported here differs in these respects. Death was due to enterocolitis unrelated to the congenital abnormality.     

Pulmonary sequestration in older child and in adults.

Pulmonary sequestration is often found in children with recurrent pneumonia, but is not rare in adults. During the last 6 years (1985-1990), 6 patients with pulmonary sequestration were surgically treated. One 40-year-old patient had an extralobar sequestration, and 5 patients ranging in age from 14 to 26 years had an intralobar sequestration. The patient with extralobar sequestration was asymptomatic. In cases of intralobar sequestration, 4 patients were symptomatic. All 6 patients had an abnormal chest roentgenogram. Preoperative arteriography revealed anomalous blood supply came from a systemic artery in 4 patients. All cases with intralobar sequestration had a lower lobectomy, and one case with extralobar sequestration had an extirpation. Even in cases without any symptoms, there was the possibility of a microscopic infection in the sequestrated lesion with the occurrence of later symptoms. Therefore, patients diagnosed with pulmonary sequestration should be considered for surgical resection.