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.     

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 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.     

One stage operation for aneurysm of the diverticulum of the ductus arteriosus and coronary artery bypass grafting

Aneurysm of the diverticulum of the ductus arteriosus in the adult is rare. One stage operation for aneurysm of the diverticulum of the ductus arteriosis and coronary artery bypass grafting (CABG) is reported. A 61-year-old man was admitted for diagnosis of thoracic aneurysm on chest X-ray and CT. Chest CT scan showed an aneurysm above the left main pulmonary artery. An aortography showed the left vertebral artery originated directly from the aortic arch and a saccular aneurysm arising from the aortic isthmus and lesser curvature of the aortic arch. Coronary arteriography showed 75% stenosis at the right coronary artery (seg. #1) and 75% stenosis at the left anterior descending artery. Operation was performed through a median sternotomy. The aneurysm of 6 to 3 cm was located between the aortic isthmus and left pulmonary artery. Ascending aorta and right atrium were used to institute cardiopulmonary bypass (CPB). CABG (LITA to #7, SVG to #4 PD) was performed. Arterial cannulation was then switched to the left femoral artery. The proximal aorta was cross-clamped between the left vertebral artery and the left subclavian artery under the partial CPB, and the distal aorta was occluded with a occulusive balloon catheter via the right femoral artery. The selective left axillar artery cannulation was performed to perfuse LITA. The aneurysm was resected and closed with a patch. His post-operative 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.     

Single coronary artery with bicuspid aortic valve stenosis and aneurysm of the ascending aorta: report of a case

A 73-year-old man with a severely stenosed bicuspid valve and an aneurysm of the ascending aorta underwent valve and aortic surgery. Preoperative imaging revealed a single coronary artery arising from the right side of the sinus of Valsalva and a branch that perfused into the left side of the heart to pass through the front of the pulmonary artery. We replaced the aortic valve and ascending aorta, painstakingly avoiding damage to the coronary artery and obstruction of the sole coronary ostium.     

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.     

Thoracoscopic segmentectomy: one vessel may hide a second one

We report the case of an asymptomatic neonate prenatally diagnosed with a left basal pulmonary sequestration. The preoperative chest computed tomography with contrast showed 2 aberrant arteries arising from the distal thoracic aorta and supplying the intralobar left inferior lung malformation. Strategy and treatment by thoracoscopic segmentectomy are presented.     

A ruptured thymic branch aneurysm mimicking a ruptured aortic aneurysm,with associated bronchial artery aneurysms: Report of a case

A 57-year-old woman who went into shock following an acute left hemothorax was operated on after stabilization under the diagnosis of a ruptured aortic aneurysm. A left fifth intercostal thoracotomy was done which revealed approximately 500 ml of bloody effusion in the extrapleural space and 2,000 g of clotted blood in the pleural cavity. While the aneurysm was initially thought to have originated in the isthmic or descending aorta, intraoperative findings revealed a swollen hematomatous thymus adherent to the aorta. A ruptured thymic branch aneurysm, 3 cm in diameter, was subsequently found in the resected hematomatous thymus. Histological examination also revealed several small aneurysms in the tortured bronchial arteries. Postoperative angiography showed a saccular aneurysm, 1.5 cm in diameter, and several smaller aneurysms in the bronchial artery of the left lung. The aneurysm was successfully treated by a transcatheter arterial embolization, and the patient has had no further symptoms since then. To our knowledge, there has been no other case of a ruptured thymic artery aneurysm reported in the literature, and only a few cases of bronchial artery aneurysms have been documented.     

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.     

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.     

Surgical treatment for cervical aortic arch with aneurysm formation

Cervical aortic arch is an unusual malformation. Cervical aortic arch with aneurysm formation is very rare. We report a case of cervical aortic arch associated with a saccular aneurysm in a 59-year-old Japanese man. The aneurysm protruded caudally and was located between the left common carotid and left subclavian arteries. Cardiopulmonary bypass and deep hypothermic circulatory arrest was applied as adjunct methods. A Dacron graft was sutured just distal to the left common carotid artery, with the patient in the Trendelenburg position. The proximal site was left open while oxygen-saturated venous blood was supplied in a retrograde manner to perfuse the lower body during occlusion of the descending aorta. Distal anastomosis to the descending aorta was performed during rewarming. The left subclavian artery was reconstructed by using a branch of the graft. This procedure is simple and useful for distal arch operations, especially in patients with Haughton D type aneurysms.     

Surgical treatment for cervical aortic arch with aneurysm formation

Cervical aortic arch is an unusual malformation. Cervical aortic arch with aneurysm formation is very rare. We report a case of cervical aortic arch associated with a saccular aneurysm in a 59-year-old Japanese man. The aneurysm protruded caudally and was located between the left common carotid and left subclavian arteries. Cardiopulmonary bypass and deep hypothermic circulatory arrest was applied as adjunct methods. A Dacron graft was sutured just distal to the left common carotid artery, with the patient in the Trendelenburg position. The proximal site was left open while oxygen-saturated venous blood was supplied in a retrograde manner to perfuse the lower body during occlusion of the descending aorta. Distal anastomosis to the descending aorta was performed during rewarming. The left subclavian artery was reconstructed by using a branch of the graft. This procedure is simple and useful for distal arch operations, especially in patients with Haughton D type aneurysms.     

A thoracic aortic aneurysm with respiratory failure caused by the mechanisms of stenosis of the left bronchus and ipsilateral pulmonary artery--report of a case and review of the literature

Clinical symptoms of thoracic aortic aneurysm vary widely from symptoms resulting from its rupture to those due to compression of surrounding organs. Furthermore, the onset of symptoms is frequently followed by a fatal outcome unless emergency measures are taken. We have recently experienced a patient who developed respiratory insufficiency caused by an aneurysm of the descending thoracic aorta compressing the left primary bronchus and the right pulmonary artery, the former causing ventilatory failure in the left lung and the latter interfering with blood flow in the right lung. The patient underwent emergency prosthetic graft replacement and showed temporary symptom improvement. However, a postoperative complication of esophageal perforation due to ischemic necrosis occurred and eventually caused acute empyema followed by death. The patient was a 48-year-old male who presented with symptoms similar to bronchial asthma. Angiographic examination (iv-DSA) revealed compression of right pulmonary artery by an aneurysm of the descending thoracic aorta. Bronchoscopic findings included extramural stenosis of the left bronchus accompanied by arterial pulsation. As an emergency measure, artificial blood vessel replacement of the descending thoracic aorta was performed. However, the patient developed postoperative acute empyema and died on the 19th postoperative day. Autopsy revealed ischemic necrosis on the esophageal wall where the aneurysmal lesion had touched before operation. This seems to have formed a perforation into the left thoracic cavity and ultimately to have caused acute empyema. Since we believe that the pathogenesis and clinical course observed in this patient is rather rare, the case is reported in accompanied by relevant data from the literature.     

Thoracic aneurysm expansion secondary to collateral supplies via thyrocervical trunk and intercostal arteries in a patient after thromboexclusion.

A case of thoraco-abdominal aortic aneurysm complicated after permanent clamping of the descending aorta (thromboexclusion) is reported. Angiographic and operative findings were: (1) a pseudo-aneurysm right at the distal anastomosis of previous intrathoracic bypass for pseudo-coarctation of the aorta filled by left ninth intercostal artery, which was supplied by the left internal thoracic artery; and (2) the cervical and thoracic spinal cord were supplied by the left vertebral artery and the mediastinal branch of the left thyrocervical trunk. This rare cause of a thoraco-abdominal aortic aneurysm and the significance of the subclavian artery as a source of spinal cord blood supply are discussed.     

Surgical treatment for ruptured aneurysm of a coronary artery to pulmonary artery fistula; report of a case

We reported a successful operative case of ruptured coronary artery aneurysm associated with coronary artery to pulmonary artery fistula. The patient was a 74-year-old woman who was admitted for syncope and chest oppression. Echocardiogram and chest CT scan revealed cardiac tamponade. Ruptured coronary aneurysm, 3 cm in diameter, was found at emergent operation. After emergent coronary angiography, which demonstrated an aneurysm arising from the left anterior descending coronary artery and draining into the pulmonary artery, orifice of draining artery to the aneurysm was closed and coronary artery fistula was ligated. She recovered smoothly and post operative angiogram revealed disappearance of the aneurysm.     

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.     

Right main bronchus obstruction in a patient intubated with a left double-lumen tube for thoracic aortic surgery

A 29-year-old woman with a tubular thoracic aortic graft prosthesis, placed after repair of a postcoarctation site aneurysm, was reoperated due to fungal infection of the prosthesis. A left thoracotomy was performed and single lung ventilation established through a double lumen tube followed by extracorporeal circulation (ECC), deep hypothermia, and cessation of blood circulation. The venous drainage tube was placed in the right pulmonary artery and arterial return was located in the descending aorta. After replacement of the prosthesis and interruption of ECC, attempts to ventilate the right lung failed. Fiberoptic examination revealed that the carina and tracheal wall completely occluded the opening of the right main bronchus. ECC was interrupted and the left lung was ventilated, but after withdrawal of the tube from the pulmonary artery and repositioning the patient in supine decubitus, oxygenation and right lung ventilation improved. The patient was extubated after 20 hours and discharged home 6 weeks later.     

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.