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.     

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.     

Intralobar pulmonary sequestration treated by resection of the sequestrated segment

Segmentectomy including the sequestrated segment and lobectomy are generally performed for intralobar pulmonary sequestration. We report a case of intralobar pulmonary sequestration of Pryce type III treated by resection of only the sequestrated segment. A 57-year-old man presented with a 5 x 3 cm mass shadow overlapping a cardiac shadow on the left lower lung field on chest radiograph at medical examination. The mass was not connected with the bronchial tree, and was supplied by an aberrant artery arising from the thoracic descending aorta. The mass was suspected to exist in the normal visceral pleura from chest images. The mass was suspected to be an intralobar pulmonary sequestration from the abovementioned findings, and we performed an operation. At the beginning of the procedure, after dissecting the aberrant artery, only the sequestrated segment was performed.     

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.     

Anomalous systemic arterial supply to the basal segment of the left lung; report of a case

A 35-year-old female was admitted to our hospital for hysteromyoma. Chest X-ray showed a mass shadow in the left lung field. A thorough examination revealed anomalous systemic arterial supply to the basal segment of the left lung with 2 abnormal arteries. Because the patient had no symptoms, no operation was performed. After 9 months however, the patient had bloody phlegm and chest pain and received an operation. Thus, we propose that the operation for the pulmonary sequestration is to be performed when such a diagnosis is made.     

Video-assisted thoracoscopic surgery for ligation of anomalous systemic arterial supply to pulmonary sequestration

A 52-year old female with anomalous systemic arterial supply to pulmonary sequestration was reported. The patient was admitted because of an abnormal lung shadow on chest X-ray film. Computed tomography (CT) showed an anomalous systemic arterial supply to pulmonary sequestration of the left lower lung without lung infection. Video-assisted thoracoscopic surgery for ligation of the anomalous systemic artery was performed. Postoperative course has been uneventful for 14 months after surgery. Blood supply increased to the left lower lung by 3-dimensional CT after surgery. The ligation of anomalous systemic arterial is enough for this disease.     

Subdiaphragmatico-mediastinal lipoma through the foramen of Bochdalek; report of a surgical case

A 53-year-old man was admitted to our hospital for evaluation of chest abnormal shadow at the left cardiophrenic angle. Computed tomography (CT) scan revealed a homogeneous mass of fatty density on the left diaphragm. Magnetic resonance imaging (MRI) demonstrated a well-demarcated tumor mass with a fatty signal intensity and, in the sagittal view, the mass showed continuity into the retroperitoneal fatty tissue. Extirpation of the tumor was performed under thoracoscopic procedure. The tumor was adhesive closely to diaphragm and extended into the retroperitoneal space through the foramen of Bochdalek. Pathologically the tumor was diagnosed as a mature lipoma. This tumor was considered to originated from the retroperitoneal fatty tissue under the diaphragm and to herniate into the mediastinum through the foramen of Bochdalek.     

Complete resection of thymic carcinoma supported by cardiopulmonary bypass

We would like to report on a case of thymic carcinoma that could be completely resected supported by extracorporeal circulation. A 53-year-old female complaining of severe coughing and hoarseness was admitted to our hospital. Her chest X-ray film revealed an abnormal shadow in the upper mediastinum and an elevation of the left diaphragm. Chest CT and MRI showed an anterior mediastinal mass that invaded the main pulmonary artery. The preoperative histological diagnosis of thymoma was made by mediastinoscopic specimen. After 50 Gy irradiation, surgery was performed. During the operation, after resection of the left brachiocephalic vein and a part of the upper lobe of the left lung, the tumor was detached from the wall of the aorta and resected with the invaded part of the pulmonary artery supported by cardiopulmonary bypass. Reconstruction of the pulmonary wall defect was accomplished with Xenomedica. The final pathological diagnosis was thymic adenosquamous carcinoma. The postoperative course has been uneventful 24 months after surgery. Extracorporeal circulation is a useful technique in operations involving malignant diseases when complete resection is able to be accomplished.     

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.     

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 adenocarcinoma of the fetal lung type; report of a case

An asynptomatic 58-year-old male was admitted to the hospital because of an abnormal nodule in the left lung field on screening chest X-ray. Chest computed tomography (CT) showed a tumor shadow mass in the left lower lobe. Open biopsy was performed to diagnose the mass. The cytological diagnosis was low-grade malignant adenocarcinoma, underwent left lower lobectomy. The histological diagnosis was pulmonary adenocarcinoma of the fetal lung type which was one of pulmonary blastoma (PB). Recently, the concept of pulmonary blastoma has changed. It will be useful to investigate the old PB's case reports to classify new concept.     

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.     

A case of pulmonary adenocarcinoma of arising from the wall of a giant bulla which disappeared spontaneously

In a 53-year-old man, giant bulla was detected on the left side on chest X-ray and mass shadow was demonstrated near the bullae (20 x 10 mm). CT scan did not show a mass shadow and the shadow was diagnosed as scar formed by the bulla wall. Three months later, the bulla had disappeared spontaneously, but an abnormal shadow at the apex of left lung was pointed out. Chest CT findings suggested lung cancer arising from the wall of the collapsed giant bullae and partial resection of the left upper lobe was performed. The postoperative pathological diagnosis was moderately differentiated adenocarcicoma (12 x 5 x 5 mm) arising from pulmonary bullae. It must be noted that occult cancer may exsist at the wall of giant bullae, the surgical procedure involving complete resection of the bullae wall must be selected.     

Giant pleural fibroma with an abdominal vascular supply mimicking a pulmonary sequestration

A 37 -year-old woman was found to have a giant mass in her right chest with an abnormal abdominal vascular supply at preoperative workup. Suspecting a pulmonary sequestration, the lesion was embolized and then removed through a standard thoracotomy. A broad-based, vascularized pedicle connected the mass to the diaphragm. Final pathology demonstrated a solitary fibrous tumor of the pleura with no malignant features. We believe this is the first case of solitary fibrous tumor of the pleura with a direct vascular supply from the abdominal aorta at the level of the renal arteries reported in the literature.     

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.     

Pulmonary sequestration associated with fungal infections; report of a case

A 45-year-old female was admitted to our hospital complaining of cough and purulent sputum. Enhanced chest computer tomography(CT) showed that abnormal mass shadow in left lower lobe of the lung and an aberrant artery originated from the discending aorta and penetrated the lesion. Left lower lobectomy was performed and intralobar sequestration was diagnosed. Moreover, fungus which were suspected Aspergillus were found in the cystic lesion. The postoperative course was uneventful and she was discharged.     

Lipoma of the diaphragm; report of a case

A 50-year-old man was referred to our hospital with an abnormal shadow on chest roentgenogram taken on a health examination in September, 1999. His past history was an operation for scoliosis at the age of 39 years. His family history was unremarkable. Chest roentgenogram showed a smooth rounded bulging nodule in continuity with the left diaphragm. Chest computed tomography (CT) revealed a well-defined and homogeneous nodular lesion with a low attenuation value of -100 Hounsfield unit, corresponding with a fat density, on the base of the left hemithorax. For making diagnosis and treatment, videothoracoscopic surgery was performed in January, 2000. The tumor was removed with combined resection of diaphragm, using stapling device, because the lesion partially adhered to the diaphragm. The postoperative course was uneventful. The pathological diagnosis was lipoma About 2 years later. he was again found to have a similar abnormal shadow on a routine chest roentgenogram in August, 2003. Chest CT showed a nodular lesion involving a previous operative staple line on the base of the left hemithorax. We considered this lesion to be a locally recurrent tumor and performed surgical resection of the lesion with a wide margin of normal diaphragm and partial retroperitoneal fat tissue. The pathological diagnosis of the resected specimen was again lipoma. Since then, he has been in good health without recurrence for about 4 years.     

Left pneumonectomy for lung cancer after correction of contralateral partial anomalous pulmonary venous return

We report the successful treatment of a 48-year-old man with left lung cancer and contralateral partial anomalous pulmonary venous return (PAPVR). He was found to have an abnormal shadow on a regular checkup. Sputum cytology revealed squamous cell carcinoma. Chest computed tomography showed not only a left hilar mass but also showed that his right superior pulmonary vein was draining into the high portion of the superior vena cava. In the presence of the right partial anomalous pulmonary venous return, it was believed that left pneumonectomy would cause serious postoperative heart failure due to an increase in the left-to-right shunt. Therefore his partial anomalous pulmonary venous return was corrected first under cardiopulmonary bypass, and 3 weeks later he underwent successful radical left pneumonectomy.     

A case of non-functioning adrenal cortical carcinoma with pulmonary and bony metastases

A 49-year-old woman was admitted to our hospital with complaints of edema and abnormal shadow in right pulmonary area. CT scan and abdominal aortography showed left adrenal mass. Moreover, the lung metastasis measured 60 x 65 mm and bony metastases were suspected in three areas on bone scanning. Under diagnosis of left non-functioning adrenal tumor with pulmonary and bony metastases, left adrenalectomy was performed. Histological diagnosis was left adrenal cortical carcinoma without lymph node metastasis. Treatment consisted of 3 g o,p-DDD and 400 mg carmofur per day after surgery. At present, pulmonary metastasis has decreased in size and bony metastases decreased in uptake on bone scan 12 months postoperatively.     

Pulmonary blastoma

We describe a resected pulmonary blastoma in an 84-year-old male, the oldest of previously reported patients. A chest X-ray showed a 2 cm-sized abnormal shadow in the left lung field. Five months later computed tomography demonstrated a well-demarcated heterogenous mass, measuring 12 cm in diameter, in the left lower lobe of the lung. This mass was diagnosed as a carcinoma using echo-guided percutaneous biopsy. The patient underwent a left lower lobectomy. The resected specimen revealed the tumor to be a pulmonary blastoma arising from lung tissue. Preoperative diagnosis of pulmonary blastoma is extremely difficult because of the histological heterogeneity of tumor. Since it has been noted that pulmonary blastoma rapidly progresses in a short period of time, surgical treatment should be undertaken as quickly as possible when such a tumor is suspected.