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 sequestration. A missed diagnosis

Intralobar pulmonary sequestration is an uncommon but distinct clinical entity that may be the unrecognized cause of recurrent pulmonary infections. Between 1967 and 1987, 10 patients, ranging in age from 5 to 39 years, were found to have an intralobar sequestration. Nine patients (90%) had a history of recurrent pulmonary infections, chronic cough, and intermittent fevers. One patient was asymptomatic. Many patients had been treated with antibiotics on numerous occasions. The delay in diagnosis varied between 3 months and 7 years (mean delay, 1.5 years). The chest roentgenogram was abnormal in all patients. The intralobar sequestration was present in the left lower lobe in 7 patients and the right lower lobe in 3 patients. Bronchography was abnormal in 4 patients in whom it was done. Bronchoscopy was performed in 7 patients, but it was only helpful in excluding other diagnoses. Preoperative thoracic arteriography in 9 patients visualized the systemic arterial supply from the thoracic or abdominal aorta to the intralobar sequestration and helped prevent any catastrophic surgical bleeding. A lobectomy was performed in 9 patients and a segmentectomy in 1 patient without morbidity or mortality. In patients with recurrent infections in the same lower lobe, a high index of suspicion for an intralobar sequestration should prompt early diagnostic arteriography and, if confirmed, early operative intervention.     

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.     

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 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.     

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.     

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 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.     

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.     

Coil embolization to treat pulmonary sequestration in the right upper lobe

Although there have been a few case reports of pulmonary sequestration, it is primarily located in the lower lobe and left lung, rarely in the right upper lobe. Here, we report a case presented with haemoptysis. Computed tomography images revealed flake ground-glass shadows in the right upper lobe. Computed tomography angiography demonstrated an artery supplied the affected lesions stemmed from the aortic arch. We diagnosed and treated her with bronchial artery angiography with coil embolization. No complications were found after operation until now. Thus, CTA could help identify the abnormal blood vessels, and interventional therapy may be an effective alternative to surgery of pulmonary sequestration.     

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.     

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.     

Pulmonary cystic adenomatoid malformation with anomalous vascularization with systemic origin

We report an infant with the diagnosis of Congenital Cystic Adenomatoid Malformation. He was operated and we found an abnormal systemic blood supply associated in that lobe. We make a review of the literature, and we make a distinction between this rare combination and others like pseudo pulmonary sequestration, or Congenital Cystic Adenomatoid Malformation associated to pulmonary sequestration.     

Intralobar pulmonary sequestration supplied by multiple anomalous arteries: report of a case

Pulmonary sequestration is abnormal pulmonary tissue that has separated from the normal pulmonary parenchyma, is not connected to the tracheobronchial tree, and is supplied by a systemic artery. We describe herein a case of intralobar pulmonary sequestration found in a 66-year-old man who was admitted to our hospital with hemoptysis, coughing, and fever. Angiography showed that the branches of the 11th left intercostal artery and a bronchial artery had formed a hypervascular area in the lower part of the left lung. Bronchial artery embolization and subsequent embolization of the left 11th intercostal artery were performed in an attempt to control the recurrent hemoptysis. These treatments were unsuccessful, and he was transferred to our department of surgery after coughing up about 400 ml of fresh blood. A left lower lobectomy was performed. The resected lung contained a large feeding artery, some acute and partly organizing inflammatory lesions within collapsed lung parenchyma, and massive intra-alveolar hemorrhage in the peripheral area. The patient had an uneventful recovery and was discharged 22 days after his operation. Received: March 24, 2000 / Accepted: January 9, 2001     

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.     

单操作孔胸腔镜手术治疗肺隔离症的临床体会

目的 总结单操作孔胸腔镜手术治疗肺隔离症的临床经验,评价其安全性、有效性。方法 回顾性分析2015年1月至2019年5月共21例单操作孔胸腔镜手术治疗的肺隔离症患者临床资料,其中男性10例,女性11例,年龄21~57（39.5±9.2）岁,21名患者均为下肺病变,包括左下肺13例,右下肺8例;叶外型1例,叶内型20例,术前均行增强CT扫描和三维重建;双腔气管插管全麻下单操作孔胸腔镜行肺叶或肺段切除20例,中转开胸1例。结果 胸腔严重粘连者8例,行下肺叶切除术20例,下肺后基底段切除1例;手术均顺利完成,手术时间（167.3±61.5）min,术中出血量（157.3±30.2）mL,术后带管时间（5.3±1.6）天,术后住院时间（6.2±1.5）天,围手术期无死亡及严重并发症发生;随访21例无失访,随访时间7~59个月,21例患者复查胸片或CT无复发。结论 单操作孔胸腔镜手术治疗肺隔离症安全可行,术前合理的检查评估和术中对隔离肺异常血管的合理处理是手术成功的关键。     

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.     

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.     

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.     

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.