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.     

Unique extralobar sequestration with atypical location and aberrant vessels

We report a unique extralobar pulmonary sequestration in the upper thoracic region with 3 aberrant vessels connecting to the right subclavian artery, right superior pulmonary vein, and right pulmonary artery in a 20-year-old man. The sequestered lung was completely excised using a video-assisted thoracoscopic approach.     

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 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.     

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.     

Thoracoscopic resection of intra- and extralobar pulmonary sequestration in the first 3 months of life

Background

Thoracoscopic techniques have gained increasing acceptance in pediatric surgery, but experience with newborns and small children is limited. To our knowledge, a series of minimally invasive resection of pulmonary sequestration in newborns has not yet been reported in the literature. We report on 5 patients with pulmonary sequestration thoracoscopically.

Methods

From November 2000 to November 2002, 5 patients underwent thoracoscopic resection of pulmonary sequestration. Ages ranged from 4 to 91 days. Two patients had postnatal pulmonary symptoms. Preoperative diagnosis was dubious in 4 children. There were 4 extralobar and 1 intralobar pulmonary sequestrations.

Results

Thoracoscopy was performed with 3-mm instruments and 3 to 5 ports. All procedures were completed successfully. The median duration of the operation was 95 minutes (range, 63-117 minutes), and visualization was excellent. Anomalous blood vessels were clipped and/or ligated. Four patients were extubated immediately after the operation, 1, the day after. The postoperative course was uneventful in all children. At follow-up after 14 months (mean; range, 10-19 months), all patients were free of symptoms and had normal chest x-rays.

Conclusion

Thoracoscopy is feasible for resection of intra- and extralobar pulmonary sequestrations during the first 3 months of life.     

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.     

Extralobar pulmonary sequestration in a 4-months infant

Aim-Background

To highlight the rarity of a congenital condition of Pulmonary sequestration. This condition is presenting in two forms, intralobar and extralobar.

Case report

An extralobar sequestration in a 4-month-old infant is reported. A mass located in the left lower pulmonary lobe was diagnosed by a prenatal ultrasound examination at the sixth month of the pregnancy had set the possible diagnosis of cystic adenomatoid dysplasia of the left lower pulmonary lobe. No distress or other symptoms were observed. Surgical intervention with a left posterolateral thoracotomy over the fifth intercostal space allowed approach to the sequestration. The clinical presentation, differential diagnosis and therapeutic approach are discussed herein.

Conclusions

It is significant for the medical team to consider pulmonary sequestration in the differential diagnosis for any patient that presents with respiratory distress and a chest radiograph showing a mediastinal shift.     

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.     

Malignant spindle cell tumour (fibrosarcoma) in sequestration of lung—a case report

Pulmonary sequestration is an uncommon congenital anomaly of respiratory system and usually diagnosed in childhood but can present at any at age. It frequently presents as a mediastinal mass with symptoms from vascular shunting, or associated anatomic defects. This case report describes an adult who presented with chest pain due to extralobar sequestration of right lung extending into the mediastinum. Per-operatively it was found to be an extra pulmonary mass lesion with a peduncle attached to right lung suggestive of extralobar sequestration. Histopathological examination of the specimen showed it to be spindle cell tumor (fibrosarcoma) in sequestrated lung. The patient had an uneventful recovery and is under regular follow up without any further problems.     

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.     

Pulmonary sequestration. A report of an unusual case and a review of the literature

A case of extralobar sequestration with findings indicating a pulmonary origin is reported. The literature has been reviewed and evidence presented that intra- and extralobar sequestration are variants of the same abnormality and that the systemic artery is an associated anomaly. From the evidence available at present the pathogenesis of this anomaly is best explained as an abnormality of bronchial and foregut budding resulting from failure of normal embryonic organizer control.     

Intra-abdominal pulmonary secuestration as an exceptional cause of abdominal mass in the adult

INTRODUCTIONPulmonary sequestration (PS) is an extremely rare malformation defined as a portion of lung tissue isolated from the pulmonary system. PSs are classified into intralobar type and intra-abdominal PS that represents only 2.5% of cases. There are 20 cases of PS reported in adults and only two were managed by laparoscopic approach. We report a case of intra-abdominal PS mimicking a gastroesophageal duplication cyst in an adult. Besides its rarity, this is the first intra-abdominal PS in an adult managed by an anterior laparoscopic approach.PRESENTATION OF CASEA 60-year-old female patient had had epigastric and left upper quadrant pain for several days. Physical examination was normal. Image test were consistent with a gastroesophageal duplication. The patient was taken to the operating room for laparoscopic exploration and resection. The pathological diagnosis was extralobar pulmonary sequestration.DISCUSSIONLess than 20 cases of PS have been reported in adults and only two cases were managed by a lateral laparoscopic approach. In contrast to these reports, we used an anterior approach due to the GEJ suspected origin of the mass.CONCLUSIONExtralobar intra-abdominal PS is an extremely rare condition during adulthood but this diagnosis should be included in the differential diagnosis of a left-sided suprarenal mass. Due to the difficulty in achieving a definitive preoperative diagnosis, surgery is recommended. Laparoscopic resection is safe and effective but careful preoperative imaging studies are recommended in order to plan the most suitable approach.     

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.     

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)     

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.     

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.     

Video-assisted thoracoscopic resection for intralobar pulmonary sequestration

Video-assisted thoracoscopic treatment of intralobar pulmonary sequestration in an obese (120 kg, body mass index 42) young man is described. Two aberrant arteries originating from the descending thoracic aorta were transected with a vascular stapler, and a left lower lobectomy was successfully performed thoracoscopically. Three-dimensional chest computed tomography was used to identify the aberrant vessels. Video-assisted thoracoscopic lobectomy is useful and minimally invasive for treating pulmonary sequestration, especially in obese patients.     

Infarction of extralobar pulmonary sequestration after blunt trauma

Pulmonary sequestration is a rare congenital lung malformation that occurs in either an intralobar or extralobar location and is generally asymptomatic. We report an unusual case of a child who presented with infarction of a previously asymptomatic extralobar pulmonary sequestration after blunt trauma, eventually requiring surgical excision.