Systemic arterial supply to the left basal segment without the pulmonary artery: four consecutive cases.

Systemic arterial supply from the descending thoracic aorta to the basal segment of the left lower lobe without a pulmonary artery supply is a rare congenital anomaly within the spectrum of pulmonary sequestration cases. We encountered four consecutive cases, which were treated successfully by three basalectomies and one lower lobectomy to preserve lung function.     

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.     

Anomalous systemic arterial supply to normal basal segment of the left lower lobe; report of a case

A 28-year-old male was referred to our hospital because of hemoptysis. A chest X-ray revealed an increase of vascular marking in the left lower field and a partial defect in the lateral line of the descending thoracic aorta. An aortogram and pulmonary arteriogram showed a large artery arising from the descending thoracic aorta and supplying the left basal segment, which had no normal pulmonary arteries. A bronchoscopy showed no abnormal findings in the bronchial tree. A clinical diagnosis of systemic arterial supply to the basal segment of the left lower lung was made, and a left lower lobectomy and closure of the anomalous systemic artery by video-assisted thoracic surgery (VATS) were successfully performed. Vascular marking of the visceral pleura of left lower basal segment was observed and the anomalous arterial pressure was 84 mmHg, as high as systemic arterial pressure, during the procedure. The histopathological examination revealed normal alveolar structure, and sclerosis and hypertrophy of pulmonary arteries of the lesion (Heath-Edwards V, which means irreversible vascular changes due to pulmonary hypertension). The patient had an uneventful postoperative course and was discharged on postoperative day 8. The VATS procedure is a more useful and less invasive method for cases of systemic arterial supply to the basal segment of the left lower lung than an open thoracotomy.     

A case of pryce type I intrapulmonary sequestration

A twenty-year-old asymptomatic man hospitalized because of a vascular murmur and abnormal shadow in the left lower lung on X-ray film. An aortogram revealed an abnormal artery arising from the descending thoracic aorta and supplying the left basal segment, which had no other pulmonary arteries. Although lung ventilation scintigraphy demonstrated reduced ventilation to the left lower lobe, bronchogram showed an almost normal bronchial tree except that peripheral branches were slightly thin. A clinical diagnosis of Pryce type I intrapulmonary sequestration was made, and left lower lobectomy was performed successfully. We have analyzed 31 cases of Pryce type I intrapulmonary sequestration in Japan. A vascular murmur is often heard, and a chest X-ray usually shows either a mass shadow or increased vascular markings. In most of those cases, an abnormal artery arises from the descending thoracic aorta and it supplies the left basal segment. Because this type of sequestration causes hemoptysis and infections, surgical intervention is indicated.     

Large anomalous systemic arterial supply to basal segments of the left lung

A 29-year-old man with a large anomalous systemic arterial supply to basal segments of the lower lobe was reported here. Pulmonary arteriography showed the absence of blood flow to basal segments. Aortography verified the anomalous artery from the descending thoracic aorta. Lobectomy was performed because the anastomosis between the anomalous and pulmonary arteries was anatomically difficult and segment 6 was small. The resected specimen showed that the anomalous artery had findings of pulmonary hypertension equivalent to grade V in the classification of Heath and Edwards. If the anastomosis between the anomalous and pulmonary arteries is anatomically feasible, pathological examination of open lung biopsy specimen is necessary to determine the operative procedures in this disease and two-staged operation is desirable.     

Anomalous systemic arterial supply to the basal segments of the left lung with two aberrant arteries

A 30-year-old man was admitted to our hospital because of hemoptysis. Digital subtraction aortography revealed an anomalous systemic artery (10 mm diameter) from the descending thoracic aorta to the basal segments of the left lung. The presence of another smaller aberrant artery from the abdominal aorta was strongly suspected on the basis of aortography. We confirmed the presence of a smaller aberrant artery (3 mm diameter) traversing the pulmonary ligament after thoracotomy. We performed left lower lobectomy with resection of the two aberrant arteries via posterolateral thoracotomy with a favorable postoperative outcome. Although it is rare, the possibility of the presence of several aberrant arteries should be considered in anomalous systemic arterial supply to the basal segment of the left lung. Preoperative identification of aberrant arteries was useful for a safe operative procedure.     

Anomalous systemic arterial supply to the basal segments of the left lung with two aberrant arteries.

A 30-year-old man was admitted to our hospital because of hemoptysis. Digital subtraction aortography revealed an anomalous systemic artery (10 mm diameter) from the descending thoracic aorta to the basal segments of the left lung. The presence of another smaller aberrant artery from the abdominal aorta was strongly suspected on the basis of aortography. We confirmed the presence of a smaller aberrant artery (3 mm diameter) traversing the pulmonary ligament after thoracotomy. We performed left lower lobectomy with resection of the two aberrant arteries via posterolateral thoracotomy with a favorable postoperative outcome. Although it is rare, the possibility of the presence of several aberrant arteries should be considered in anomalous systemic arterial supply to the basal segment of the left lung. Preoperative identification of aberrant arteries was useful for a safe operative procedure.     

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.     

Abnormal systemic artery originating from descending aorta to normal basal segments of left lung. Report 2 cases

Two patients with a large abnormal systemic artery originating from the descending aorta several centimeters above the diaphragm to the four basal segments of normal left lower lobe (without cystic change like that found in the bronchopulmonary sequestration) were treated. No pulmonary artery was found to supply the basal segments. The patients suffered from repeated hemoptysis. In one patient the abnormal artery was incidentally found during bronchial arteriography. Lower lobectomy was performed in the two patients. Microscopic examination of specimens revealed some dilated small blood vessels with extremely thin walls and their rupture may be the cause of hemoptysis. Abnormal systemic artery must be suspected if hemoptysis or local murmur during chest examination cannot be explained by other conditions, and care must be taken not to injure the artery in isolating pleural adhesion and pulmonary ligament.     

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 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.     

A case of aberrant left pulmonary basal aneurysm arising from the descending aorta]

A 65-year-old man was referred to our hospital because of fever and abnormal shadow in the left lung. An aortogram revealed a large artery arising from the descending aorta supplying the left basal segment and, which then flowed into the pulmonary vein. This artery had an aneurysm and 30 mm in diameter. Bronchography showed compression of the left basal branch. A clinical diagnosis of an aberrant left pulmonary basal aneurysm arising from descending aorta was made, and left lower lobectomy was performed, because of the danger apprehension of the aneurysmal rupture. The procedure was successful. There are only 15 case reports of a systemic artery supplying the lung with normal bronchial branch in Japan. Differences of this anomaly from pulmonary sequestration were discussed.     

A case of anomalous systemic arterial supply to the left basal lung with lateral chest pain and palpitation followed by left basal segmentectomy

A 38-year-old man was admitted because of left lateral chest pain and palpitation. A further examination revealed anomalous systemic arterial supply to the left basal lung. As pulmonary arteriography showed a complete lack of pulmonary arterial supply to these segments, we performed a ligation of the aberrant artery and left basal segmentectomy. Eight months after surgery, a lung perfusion scan showed improved uptake in the apical segment of the lower lobe.     

A case of systemic origin of an aberrant artery to the basal segments of the left lung

A thirteen-year-old asymptomatic boy was referred to our hospital because of a cardiac murmur detected on a routine physical examination at school. A plane chest radiography showed increased vascular markings in the left lower field. An Aortogram revealed a large artery arising from the descending aorta and suppling the left posterior basal segment which had no pulmonary arteries. A bronchogram showed no abnormal findings in the bronchial tree. A clinical diagnosis of systemic origin of an aberrant artery to the basal segments of the left lung was made, and left lower lobectomy was performed successfully. We discussed the difference of this anomaly from pulmonary sequestration as well as the surgical procedure.     

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.     

Anomalous systemic arterial supply to normal Basal segments of the left lower lobe of the lung: therapeutic implication

We describe a 5-month old infant who presented with a continuous murmur and enlargement of the left heart. The patient's diagnosis was an anomalous systemic arterial supply to basal segments of the left lower lobe characterized by a lack of a pulmonary arterial supply. This condition was treated without lobectomy. To our knowledge, this report is the first to describe an anomalous systemic arterial supply to basal segments of the lower lobe of the left lung with a single arterial supply that was treated in childhood without lung resection. Our case offers an alternative treatment to surgical lobectomy for this abnormality.     

Intralobar pulmonary sequestration

Sequestration is defined as an area of abnormal pulmonary tissue not connected with the bronchial tree, supplied by an aberrant systemic artery and without a normal pulmonary function. Extralobar (ELS) and intralobar (ILS) forms are distinguished. During the year 2002 the authors diagnosed and operated upon two cases of the intralobar form of pulmonary sequestration, and in last 25 years five cases - 4 x ILS and 1 x ELS. Reported are a 35 year old man with relapsing infections of the sequester and a 21 year old woman where the sequestration was accidentally found without clinical symptoms. The focus was localized in both cases in the left lower lobe of the lungs, anomalous supply arteries derived from the thoracic aorta. Venous drainage of the sequester was different - in the man a systemic drainage via the v. azygos, in the woman via the pulmonary veins was found. In one case the diagnosis was made on the basis of angiography and computer tomography, in the other case it was made on the basis of multidetector CT angiography (MDCTA). Both findings were treated by primary surgical intervention lobectomy. The postoperative course was uneventful.     

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.     

A rabbit model of highkinetic one-sided pulmonary hypertension induced by Pott's operation

This article presents a new rabbit model of pulmonary hypertension using rabbits. The left pulmonary artery of the rabbits was anastomosed on the descending aorta side-to-side and the proximal left pulmonary was banded. A portion of the arteries in the left upper lobe was ligated, and the majority of shunted blood was driven to the left middle and lower lobe. The left pulmonary hypertension was established in 86.7% of the rabbits within 1-3 months post operation and the left pulmonary hypertension was formed in all rabbits after 3 months post operation with pathological changes of pulmonary arteriole. The left pulmonary arterial pressure was higher than right pulmonary arterial pressure and the pathological changes were less evident in the right pulmonary arteriole.     

Pulmonary sequestration with high levels of tumor markers tending to be misdiagnosed as lung cancer

A 62-year-old man with hemoptysis and an abnormal shadow on chest roentgenogram was diagnosed as having anomalous systemic arterial supply to the normal basal segment of the left lower lobe. The preoperative serum carbohydrate antigen 19–9 and carcinoembryonic antigen levels were 73.8 units/ml and 10.8 ng/ml, respectively. Histopathological examination confirmed that the lesion was an intralobar pulmonary sequestration without air connection. There was no malignant finding in the resected specimen. The serum values of tumor markers returned to their approximate normal ranges after lower lobectomy.