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.     

Spectrum of Pulmonary Sequestration

Bronchopulmonary sequestration was diagnosed in 17 patients ranging in age from newborn to 64 years. The sequestration was intralobar in 14 patients and extralobar in 3. The spectrum of symptoms could be divided into three patterns: no symptoms (6 patients), respiratory problems (8 patients), and cardiovascular problems (3 patients). Cardiovascular problems usually manifest themselves in the first few weeks or months of life and often have a respiratory component. In older patients the sequestration is first manifested by recurrent pulmonary infections or, if it remains uninfected, an asymptomatic density on chest roentgenogram.The definitive diagnostic study is arteriography. Operative treatment for the intralobar variety consists of segmental resection or, if the inflammatory process is more extensive, lobectomy. An extralobar sequestration may simply be excised.     

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.     

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.     

Diagnosis and management of congenital cystic disease of the lung in children

Pulmonary sequestration, congenital adenomatoid malformation (CAM), congenital lobar overinflation (CLO), and bronchogenic cysts are four congenital lesions that may present as abnormal cystic areas within the pleural cavity in early life. They share similar clinical and embryologic characteristics, are frequently difficult to diagnose, and all require surgical treatment. From December 1974 to January 1985, 22 patients were operated upon on the Pediatric Surgical Service for congenital cystic disease of the lung. There were eight females and 14 males, ranging in age from 1 day to 18 years; 12 were under 6 months of age, and 14 were under one year. There were 7 CAMs, 5 extralobar pulmonary sequestrations, 5 intralobar pulmonary sequestrations, 3 bronchogenic cysts, and 3 cystic lobes caused by CLO. One patient had bilateral pulmonary sequestrations, intralobar on one side and extralobar on the other, with both sharing a common systemic artery arising from the infradiaphragmatic aorta. All except two asymptomatic patients presented with either progressive respiratory distress or recurrent pulmonary infections, and had cystic changes noted on chest roentgenograms. The differentiation of these congenital lesions from resolvable inflammatory cysts is important. Diagnosis was aided by selective utilization of barium contrast studies, ultrasonography, computed tomography, and arteriography. Operation consisted of resection alone of the extralobar pulmonary sequestrations and bronchogenic cysts, and total lobectomy in CAM, intralobar sequestration, and CLO. One infant with CAM died 1 day postoperatively from bilateral hypoplastic lungs. The other 21 patients are alive and well with follow-up ranging from 1 month to 9 years, with a mean of 3.5 years.(ABSTRACT TRUNCATED AT 250 WORDS)     

Infant with bilateral pulmonary sequestrations with portal venous drainage excised by video-assisted thoracic surgery

Introduction

Bilateral pulmonary sequestrations are rare congenital anomalies. Despite its benign nature, the potential complications of pulmonary sequestration (PS) are significant, including recurrent pulmonary infections, hemoptysis, congestive heart failure, and malignant potential. Therefore, the main treatment is surgical excision, even for patients with asymptomatic PS.

Case

We present an infant in whom an intralobar PS of the right lung and an extralobar PS of the left lung were diagnosed on prenatal screening ultrasonography. Both were found to have venous drainage into the portal vein. Surgical excision was performed via video-assisted thoracic surgery (VATS) at 14 months of age.

Conclusion

PS may present with unique vascular connections, including venous drainage into the portal vein. VATS resection for pulmonary sequestration is feasible and effective as an alternative to bilateral thoracotomies, in the setting of extensive preoperative planning and performance by an experienced thoracoscopic surgeon.     

Management of penetrating lung injuries in civilian practice

Recent reports of military thoracic injuries have advocated early thoracotomy and aggressive management of pulmonary injuries with resection as opposed to the more conservative and traditional treatment with chest tube thoracostomy. A retrospective study was therefore performed to determine the incidence of thoracotomy and lung resection in civilian injuries and to evaluate the effectiveness of treatment of these injuries. Between 1973 and 1985, in a series of 1,168 patients, there were 384 gunshot wounds and 784 stab wounds to the thorax. Two hundred eighty-three patients with a gunshot wound (74%) and 602 with a stab wound (77%) were treated with chest tubes alone. Sixty-eight patients (6% of the total) required operative repair of pulmonary hilar or parenchymal injury. Pulmonary resection was necessary in only 18 patients (nine with a gunshot wound and nine with a stab wound), and 10 patients had repair of hilar injuries (nine with a gunshot wound and one with a stab wound). Of patients requiring pulmonary resection, nine required wedge or segmental resection, six required lobectomy, and three patients required pneumonectomy. Mortality for all thoracic injuries was 2.3%: for those treated with chest tube alone, 0.7%; for pulmonary hilar injuries, 30%; for pulmonary parenchymal injuries, 8.6%; and for injuries necessitating lung resection, 28%. Most civilian lung injuries can be treated by tube thoracostomy alone. Although relatively few patients with primary pulmonary injury require thoracotomy, those that do are at significant risk and may require lung resection to control bleeding or hemoptysis or to remove destroyed or devitalized lung tissue.     

Pulmonary sequestration children: is the thoracoscopic approach a good option?

Background The thoracoscopic approach for pulmonary sequestration in children seems to be feasible and reproducible. To date, there have been no studies comparing the thoracoscopic approach with the conventional technique in this indication. Therefore, the aim of this study was to compare these two approachs.Methods We performed a retrospective study in patients treated surgically for a prenatal diagnosis of pulmonary sequestration between 2000 and 2003. Three or five ports were used, and single-lung ventilation was required. The clinical, operative, and postoperative data were compared with those for patients operated on during the same period via thoracotomy. For statistical purposes, the children who required conversion to the open proecedure were grouped with those who underwent thoracotomy. The following data were compared: age, and weight, operating time, duration of postoperative drainage via chest tube, duration of postoperative narcotic use, pain scores on the 1^st and 2^nd days after the procedure, and length of hospital stay. The Mann-Whitney test was used for statistical analysis.Results Five infants were operated on via a thoracoscopic approach. Two required conversion to an open procedure. There were four intralobar sequestrations (ILS) and one extralobar sequestration (ELS). The mean age at surgery was 6.2 months (range, 4–9). The mean operating time, excluding the patients who were converted to open, was 155 min (range, 120–190). In the thoracotomy group, there were one ELS and one ILS. Two groups were defined: the exclusively laparoscopy group (EL) (n = 3) and the thoracotomy and conversion group (TC) (n = 4). The duration of postoperative drainage, duration of postoperative narcotic use, and hospital stay seemed to be shorter in the group EL. These times, respectively, were 3.4 vs 2.75 days, 4.1 vs 3.3 days, and 7.5 vs 4.3 days = (p = 0.03). There were two complications in the TC group (one wound abcess and one pleural leak that required a chest tube).Conclusion The thoracoscopic approach for pulmonary sequestration is feasible. It seems to reduce the duration of postoperative drainage, postoperative narcotic use, hospital and stay. These results need to be confirmed by a prospective and more detailed study.     

Pulmonary sequestration presenting as a massive haemoptysis in adult: A case report

IntroductionPulmonary sequestration is a rare congenital lung malformation characterized by non-functioning mass of pulmonary tissue that lacks normal communication with bronchial tree and receives one or more aberrant systemic arterial supply. It usually presents in children with recurrent chest infections. It is uncommon in adult and remains asymptomatic or present with recurrent chest infections, rarely with more severe symptoms like a massive haemoptysis as in seen in our case.Case presentationA 22-year-old male presented with a chief complain of multiple episodes of massive haemoptysis. After evaluation with chest x-ray, CT chest and angiogram, he was diagnosed to have intralobar pulmonary sequestration. He underwent successful thoracotomy and left lower lobectomy. He is asymptomatic after one year of follow up.DiscussionPulmonary sequestration is rare in adult and can present with various symptoms like chest pain, cough, sputum production, recurrent infection and rarely haemoptysis. Intralobar sequestration of left lower lobe is the most common as seen in our case. The diagnosis can be made by Computed Tomogram chest with angiogram. Surgical intervention is the definitive curative treatment. Post-operative outcome is excellent with early patient satisfaction and promising outcome in long term follow-up.ConclusionBronchopulmonary sequestration can rarely present in adults presenting with massive haemoptysis. CT chest is the best modality for diagnosis. Early surgical intervention is definitive treatment with good long term outcome.     

Upper thoracic extralobar pulmonary sequestration with anomalous blood supply from the subclavian artery

The authors report on a newborn boy with extralobar pulmonary sequestration in the right upper thoracic region. Preoperative angiography showed an anomalous large vessel from the right subclavian artery, supplying the sequestrated lobe. Right thoracotomy was performed to resect the sequestrated lobe, which was diagnosed as extralobar form. This is an extremely rare case of extralobar pulmonary sequestration in which anomalous blood supply from the subclavian artery was seen preoperatively on radiographs. The authors recommend angiographic examination, particularly in case of diagnostic difficulty.     

Management of antenatally diagnosed pulmonary sequestration associated with congenital cystic adenomatoid malformation

BACKGROUND: Sequestration with associated cystic adenomatoid malformation is rare. A study was undertaken to determine whether pulmonary sequestration associated with congenital cystic adenomatoid malformation has a more favourable natural history than that of sequestration without associated cystic adenomatoid malformation. METHODS: An outline of the postnatal work up leading to the management of extralobar or intralobar pulmonary sequestration with congenital cystic adenomatoid malformation diagnosed antenatally as pulmonary malformation is presented and the indications for surgical intervention are discussed. RESULTS: In five infants in whom an antenatal ultrasound scan had detected a congenital lung malformation at 18-19 weeks gestation a final diagnosis of extralobar or intralobar pulmonary sequestration with congenital cystic adenomatoid malformation was made postnatally. Postnatal ultrasound and computerised axial tomographic scans confirmed the diagnosis of sequestration by delineating anomalous vascular supply. Cystic changes were also observed in the basal area of the sequestration in all patients. Four children remained asymptomatic and one infant presented at 10 months of age with pneumonia. The mean age at surgical resection was 6.8 months (range 2-10). Histopathological examination confirmed intralobar pulmonary sequestration with associated Stocker type 2 congenital cystic adenomatoid malformation in two patients and extralobar pulmonary sequestration with associated Stocker type 2 congenital cystic adenomatoid malformation in three patients. The mean period of follow up was four years (range 1-8). The children remain well and are developing normally. CONCLUSIONS: The importance of seeking an anomalous blood supply in children with congenital lung lesions is emphasised. Pulmonary sequestration and congenital cystic adenomatoid malformation probably share a common embryogenesis despite diverse morphology. The natural history of antenatally diagnosed lung masses is variable. Early postnatal surgical resection of pulmonary sequestration with cystic adenomatoid malformation is recommended. Surgical excision should be conservative, sparing the normal lung parenchyma.     

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.     

Pulmonary sequestration: a comparison between pediatric and adult patients.

OBJECTIVE: Modern large single institutional reports on pulmonary sequestration (PS) are extremely rare. We were interested in comparing patients with PS referred by our pediatric versus adult pulmonologists. METHODS: Hospital notes of all patients operated on between 1978 and 1997 for a congenital broncho-pulmonary malformation were reviewed. In 28 patients, the parenchymal lesion was vascularized by a systemic artery and was separated from the bronchial tree, thus matching the strict definition of PS. Patient characteristics and outcome were analyzed comparing the pediatric group (< or =16 years: n=13; mean age, 3+/-5 years) versus the adult group (>16 years: n=15; mean age, 33+/-13 years). RESULTS: No significant differences between both groups were observed in sex, side, type of sequestration, pulmonary venous drainage, associated anomalies, hospital and late outcome, and patient's overall score. Patients (n=21) with the intralobar type of sequestration presented significantly more often with an infection when compared with patients (n=7) with the extralobar type (91 versus 14%; P=0.0033). When compared with the pediatric group, patients in the adult group had significantly more respiratory infections (87 versus 38%; P=0.016), and also required a lobectomy more often (67 versus 31%; P=0.056). CONCLUSIONS: The extralobar type of sequestration often remains asymptomatic, and is usually an incidental finding during infancy. The intralobar type mostly presents with recurrent infections in adulthood resulting in more lobectomies. We believe these findings support our current policy to remove any pulmonary malformation whenever diagnosed in order to: (1), prevent infection and other potentially serious late complications which may compromise the surgical outcome; and (2), enhance the chance of a parenchymal-sparing resection.     

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.     

Techniques for Treating Aberrant Arteries During Resection of Pulmonary Sequestration by Video-Assisted Thoracic Surgery: Report of Two Cases

We describe the techniques we used for treating aberrant arteries during resection of pulmonary sequestration by video-assisted thoracic surgery (VATS) in two patients. In patient 1, the aberrant artery was transected after securing six rows of staples with a knifeless vascular endostapler. In patient 2, the aberrant artery was cut after ligation with special forceps that designed by one of us (S.K.) to push a knot, tied outside the body, into the thoracic cavity, then ligate the suture. Left lower lobectomy and right basal segmentectomy were both successfully performed by these methods. These two cases are reported to show that VATS lobectomy is a feasible and minimally invasive technique of treating pulmonary sequestration and other diseases of the lung. Received: October 12, 2001 / Accepted: July 2, 2002 Reprint requests to: S. Kaseda     

Hemoptysis and massive hemothorax as presentation of intralobar sequestration

Intralobar sequestration is a rare abnormality usually diagnosed at later age after a history of recurrent pulmonary infections. We present a case of a 55-year-old man in whom both hemoptysis and massive hemothorax were the initial presenting symptoms. This report shows that intralobar sequestration can have a dramatic course of disease, and for this reason resection of the sequestered tissue should be considered in all patients.     

Pulmonary sequestration.

Pulmonary sequestration is a congenital anomaly in which an aberrant systemic artery arising from the thoracic or upper abdominal aorta supplies part of the lungs, usually the lower lobe. The sequestered lung may be anatomically distinct from the remainder of the lobe (extralobar), or may be included in the substance of the lobe, in which case it may or may not have bronchial communication with the rest of the bronchial tree. The patients present, often in the first two decades of life, with recurrent and severe bronchopulmonary infections. Associated anomalies are present, especially in the extralobar variety. Nine cases of sequestration are reviewed, stressing significant clinical, radiological, and arteriographic findings. Preoperative demonstration of the anomalous vessel by aortography has contributed significantly to the planning and safety of the surgical procedure, which was generally a lower lobectomy. Eight out of nine patients survived the procedure.     

Hybrid minimally invasive treatment of intralobar pulmonary sequestration: a single-centre experience

Pulmonary sequestrations are rare congenital malformations. They are often located in the lower lobes, and they are supplied by an aberrant systemic vessel arising from the thoracic aorta or abdominal arteries. These pulmonary malformations are divided into intra- and extralobar sequestrations, depending on the respective lack or presence of an independent pleural covering. Pulmonary sequestration can be asymptomatic or lead to recurrent pulmonary infections. The goal of this study was to analyse the feasibility and safety of a hybrid sequential approach. We report a small series of intralobar pulmonary sequestrations, from November 2017 to December 2018, successfully treated with a hybrid minimally invasive approach consisting of endovascular embolization of the aberrant arterial branch followed by video-assisted thoracoscopic lobectomy the day after. Thoracic pain following endovascular embolization was noted in all cases. Patients were discharged early in the absence of major postoperative complications. Prolonged air leak was observed in only 1 case. Despite the presence of sequestration-related pulmonary inflammation, in our experience, hybrid treatment for intralobar pulmonary sequestration is a safe and reproducible approach in terms of postoperative complications and hospital stay.     

Emergency surgical intervention after unsuccessful percutaneous transluminal angioplasty and stenting of aortic coarctation

Two adult patients were diagnosed with extralobar and intralobar pulmonary sequestration. One patient presented with haemoptysis. Both patients suffered from recurrent episodes of severe pulmonary infections. Both patients were treated by means of endovascular embolization using Amplatzer(?) vascular plugs (AVPs). They were discharged from hospital after 48 and 24 h and then followed up for 24 and six months, respectively. No recurrence of symptoms was observed. Computed tomography scans were obtained every six months. Persistent occlusion of vascular supply and moderate regression of the sequestered lung tissue are evident after 24 and six months in both patients. Just one case of an adult patient affected by pulmonary sequestration and treated by endovascular embolization has been reported to date. The present report is the first on the use of the AVPs in adults for this condition. The potential advantages and drawbacks of this treatment modality in adults are discussed, as well the specific benefit represented by the AVPs.     

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.