Pulmonary sequestration complicated by anomalies of pulmonary venous return

Five anomalies of pulmonary venous drainage were seen among 12 children operated for lung sequestration. In two children, venous drainage from the sequestrated lobe and the rest of the right lung was via a single channel into the inferior vena cava ("scimitar syndrome"). In one of these children, the sequestrated lobe was resected and repair of the scimitar syndrome was delayed; in the second patient, the anomalous pulmonary venous drainage was not recognized preoperatively and the vein was ligated, resulting in acute hemorrhagic infarction of the right lung and death of the patient. Three patients had less severe anomalies of pulmonary venous drainage. We recommend very careful evaluation of patients with lung sequestration with special reference to pulmonary venous drainage.     

Intra-abdominal pulmonary sequestration: a case report and literature review

Pulmonary sequestration is a rare congenital malformation mostly located in the thorax, while intra-abdominal pulmonary sequestration is an extremely rare type of pulmonary sequestration usually diagnosed during the first 6 months of life. Only 1 case of intra-abdominal pulmonary sequestration in a patient older than 60 years has been reported in the current literature. It is difficult to differentiate an intra-abdominal pulmonary sequestration from other retroperitoneal tumors. A definitive diagnosis is always made by histological examination. Intra-abdominal pulmonary sequestration commonly responds well to surgical resection and is associated with excellent results and prognosis. The authors present the case of a 74-year-old asymptomatic man with a retroperitoneal mass which was completely excised and revealed by histopathological study to be an intra-abdominal pulmonary sequestration.     

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.     

Early posttraumatic pulmonary platelet trapping and its potentiation by oral pretreatment with alcohol

Soft tissue trauma is associated with platelet aggregation and sequestration in the lungs. This is believed to be an early step in the later development of adult respiratory distress syndrome. In the present experiment using a new method for in vivo dynamic studies of platelet sequestration, we wanted to evaluate the effect of soft tissue trauma on pulmonary platelet trapping in pigs and the influence of acute alcohol intoxication. The results show that significant pulmonary platelet trapping is registered within minutes of trauma and that alcohol significantly increases platelet sequestration in the lungs. This indicates an increased risk for posttraumatic pulmonary problems in alcohol-intoxicated trauma victims.     

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.     

Intralobar pulmonary sequestration and mediastinal bronchogenic cyst.

A patient with a bronchogenic cyst and intralobar pulmonary sequestration is presented. Chest radiography showed a posterior mediastinal mass and a computed tomographic scan of the chest revealed, in addition, an abnormality suggestive of pulmonary sequestration. This was confirmed by an aortogram. It is important to be aware of the coexistence of these anomalies to make a diagnosis preoperatively.     

肺隔离症4例诊治体会及文献复习

目的 探讨肺隔离症的诊断和治疗策略。方法 2017年7月至2019年6月收治的4例肺隔离症患者,3例患者行胸腔镜手术治疗,1例行介入下血管塞封堵迷走供血动脉,对其诊治过程和临床资料进行回顾性分析,总结诊治过程中的体会。结果 4例肺隔离症患者均接受治疗,1例患者接受介入下血管塞封堵迷走供血动脉后恢复顺利;1例患者胸腔镜下肺叶切除术后恢复顺利;2例患者接受胸腔镜下隔离肺叶切除术,其中1例恢复顺利,1例因胸腔进行性出血行胸腔镜下开胸止血术,后恢复顺利。术后3月复查胸部螺旋计算机体层摄影血管造影显示,3例行胸腔镜下隔离肺叶或肺叶切除手术患者的异常肺叶及供血动脉消失,行介入下血管塞封堵异常血管的1例患者的迷走供血动脉被栓塞,栓塞血管远端已无血流供应,隔离肺组织充血表现较前明显好转,4例患者随访7~31个月,未见复发。在手术时间、术中出血量、术后疼痛、术后胸腔闭式引流量、术后并发症、住院时间及住院费用等方面,介入栓塞治疗均优于胸腔镜手术治疗。结论 胸腔镜手术是目前处理肺隔离症的主要方式,介入栓塞治疗肺隔离症同样是一种安全、有效、微创的治疗方法,尤其对以咯血为主要症状,凝血功能异常且病情较重者效果佳。     

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.     

Simultaneous intra- and extralobar lung sequestration and its intermediate stages

We operated upon a patient with a simultaneous intra- and extrapulmonary sequestration in relationship to the same left lower lobe. The rarity of this form of pulmonary sequestration and its contribution to the comprehension of the pathogenesis of this malformation justifies the present publication. Most of the distinctive characteristics between the two different sequestration form found in the literature can be questioned. The unit of the lesion in our patient with the stalk connected intra- and extrapulmonary part, the identical histology, the arterial supply and venous drainage they have in common, points impressively to one single coherent structure - an organoid.     

Colonisation with Aspergillus of an intralobar pulmonary sequestration.

Pulmonary sequestration is a term used to describe an area of embryonic lung tissue supplied by an anomalous systemic artery. Two forms are recognised-extralobar and intralobar-with different clinical presentations. A patient is reported with intralobar pulmonary sequestration in the left lung and colonisation with Aspergillus which was successfully treated by lower lobectomy.     

Focal segmental glomerulosclerosis and partial deletion of chromosome 6p: a case report

We treated a patient with 6p partial deletion syndrome diagnosed after proteinuria was detected during developmental examination 3 years after birth. External anomalies included ocular hypertelorism, saddle nose, elongated philtrum, tent-like lips, and low-set auricles. Mental retardation was evident. The karyotype was 46,XX,del(6) (p.22.1-p22.3) with an interstitial deletion. The kidneys showed no abnormality on imaging such as hydronephrosis, atrophy, or malformation. Examination of a renal biopsy specimen disclosed focal segmental glomerulosclerosis. No cardiac anomaly or Rieger anomaly, which often are present in this syndrome, were noted.     

Utility of preoperative visualization for intrapulmonary sequestration in video-assisted thoracoscopic surgery

We herein report the case of a 59-year-old male suffering from severe cough. The patient had previously experienced several episodes of pneumonia beginning in childhood. A three dimensional multidetector computed tomography (3D-MDCT) scan revealed pulmonary sequestration with a left gastric artery blood supply, and clearly revealed the other pulmonary vessels. The patient underwent video-assisted thoracoscopic surgery, in which the anomalous tissue was resected safely. Preoperative visualization of the vessels may be helpful for the safe treatment of a pulmonary sequestration, and the VATS approach is a suitable operation under corrective surgical planning.     

Pulmonary sequestration with myocardial ischemia caused by vasospasm and steal

We describe patient with a rare pulmonary sequestration with myocardial ischemia in the left coronary artery caused by vasospastic angina and stealing from coronary circulation. The anterior atrial branch from the left circumflex artery gave rise to a large and anomalous vessel in the right posterior lung field. Both the surgical and medical treatment of this pulmonary sequestration improved the control of angina attacks.     

Pulmonary sequestration

A review of 41 patients over a 15-year period with a diagnosis of pulmonary sequestration was undertaken. The most common presenting complaint was repeated infections of the sequestrated segment. Two of our patients were symptomatic since the neonatal period. On an average, each patient was admitted three times to the hospital before undergoing surgery. All our patients had abnormal chest x-rays. Two patients had severe purulent infection needing emergency resection of the sequestrated lobe. In our experience, almost all cases of pulmonary sequestration require surgery in the long-term.     

Extralobar Pulmonary Sequestration Mimicking an Adrenal Tumor

Pulmonary sequestration is a rare cystic malformation composed of bronchopulmonary tissue that is discontinuous from the tracheobronchial tree and has an anomalous systemic blood supply. We present a case of a 40-y-old male who presented with an extralobar pulmonary sequestration and underwent a laparoscopic retroperitoneal mass excision. Preoperative imaging revealed a large 11.3-cm retroperitoneal tumor consisting of a multiloculated cystic lesion. The patient was discharged home, and at 3-mo follow-up no complaints were reported.     

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 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.     

An incidental and uncommon pulmonary sequestration with an uncommon feeding artery

INTRODUCTIONPulmonary sequestration is a rare congenital anomaly and most intralobar sequestrations were located in lower lobes. There is little information on middle lobe intralobar sequestration.PRESENTATION OF CASEA 44 year-old man with right middle lobe syndrome was referred for surgical management. He underwent video-assisted thoracoscopic right middle lobectomy and his postoperative course was uneventful. Pathology is notable for an incidental intralobar sequestration.DISCUSSIONOur case was unique in that the location of incidental pulmonary sequestration is uncommon and in that its feeding artery was uncommonly located in the fissure.CONCLUSIONWe report an extremely rare case of right middle lobe intralobar sequestration with a feeding artery in the fissure.     

Thoracoscopic resection of an apical extralobar pulmonary sequestration in an infant

Pulmonary sequestration is a form of bronchopulmonary-foregut malformation that is treated with surgical resection, either via a thoracotomy or the thoracoscopic approach. Apical extralobar pulmonary sequestrations are rare. We report a case of an apical pulmonary sequestration in an infant that was amenable to thoracoscopic resection. An 8-month-old girl, weighing 10.3 kg, with a left apical extralobar pulmonary sequestration underwent thoracoscopic resection. The pulmonary sequestration was identified and dissected free from surrounding tissue. The vascular peduncle was secured with an endo-GIA loaded with 2-mm vascular staples. The infant was discharged on postoperative day two without complications. In spite of the patient's small size and the apical nature of the sequestration, the operation was easily performed via a thoracoscopic approach. Thoracoscopic resection of an apical extrapulmonary sequestration in a small infant is feasible and may minimize the morbidity of this procedure.     

Endovascular treatment of symptomatic pulmonary sequestration

Pulmonary sequestration is a rare congenital malformation whose origin is bronchial and arterial simultaneously and its vascularization comes from an anomalous systemic artery. Its clinical presentation includes recurrent pneumopathy in the same anatomic location of the lung and difficult to resolve or recurrent lung abscess. It is usually treated with antibiotherapy and eventual surgical resection. A 23-year-old woman with history of recurrent respiratory infections and three episodes of hemoptysis was admitted at the hospital. Computed tomography and magnetic resonance imaging confirmed diagnosis of pulmonary sequestration. The angiographic study showed the presence of three inflow arteries arising from the thoracic aorta (T10) and supplying the abnormal lung parenchyma at the base of the left hemithorax. The patient underwent endovascular treatment consisting of exclusion of the inflow vessels with Amplatzer occlusive devices and coils. Subsequent computed tomography angiogram confirmed complete infarction of the sequestration. At 7 months, the patient presented with a new episode of bronchial infection. Repeated angiography showed persistence of intermediate small nutrient branches that were treated with coil embolization. The patient is symptom-free at 41 months after this secondary procedure. Endovascular treatment of pulmonary sequestration, with selective embolization of the inflow arteries, is a very attractive minimally invasive therapeutic option, as compared with conventional surgery, and potentially less prone to associated complications.