Pulmonary sequestration in adulthood: clinical-morphological study.

OBJECTIVES: The aim of the study was to evaluate pulmonary sequestration (PS). We report on location, blood supply, histology, clinical manifestation, and surgical treatment of PS, as well as on postoperative course in patients with PS. BACKGROUND: PS is a rare congenital defect of the lower respiratory tract, it represents locus minoris resistentiae of the body. Occasionally, PS is diagnosed for the first time in adulthood. METHODS: We evaluated 7 cases of PS treated at the Centre of Thoracic Surgery in Vysné Hágy, Slovakia, between years 2013 and 2020. RESULTS: Four of our seven patients were asymptomatic; the PS was found incidentally upon chest imaging. Three patients had recurrent bronchopneumonia related specifically to the intralobar type of sequestration. The most significant complication, observed in a singular patient, was a life-threatening episode of haemoptysis, requiring urgent surgical intervention. In the other 6 cases, the sequestra were surgically resected during the period when they were asymptomatic. and their sputum was confirmed negative upon microbiological examination. Anatomical resection of the affected pulmonary lobe by thoracotomy was the most common type of operation performed (4 cases, n = 7). There was no surgical mortality. CONCLUSION: To prevent complications, it is crucial to perform surgical treatment for pulmonary sequestration in patients who have sufficient functional capacity (Tab. 2, Fig. 4, Ref. 30). Text in PDF www.elis.sk Keywords: pulmonary sequestration, anatomic lobectomy, haemoptysis.

[The Successful Reduction of Resected Lung Volume for Intralobar Pulmonary Sequestration with Infrared Thoracoscope].

A 59-year-old male patient was referred to our hospital for further examinations and treatment due to an abnormal shadow detected in his left lower lung lobe on computed tomography. The patient was diagnosed with intralobar pulmonary sequestration and scheduled for an operation. During the surgery, after resection of the aberrant artery, indocyanine green was intravenously injected, and the border between normal lung and sequestrated lung was clearly identified by an infrared thoracoscope. Subsequently, wedge resection was performed, and the patient was discharged on postoperative day 5. Spirometry performed 6 months after the surgery indicated that the patient's lung function was well-preserved compared to the preoperative status.

[Right upper lobe pulmonary sequestration as a rare cause of recurrent spontaneous pneumothorax]. / Sekvestratsiya verkhnei doli pravogo legkogo ­ redkaya prichina pravostoronnego retsidiviruyushchego spontannogo pnevmotoraksa.

A 19-year-old patient after previous wedge resection of the right upper pulmonary lobe a year ago urgently admitted with recurrent right-sided spontaneous pneumothorax. According to standard management of spontaneous pneumothorax, we performed diagnostic thoracoscopy and drainage of the right pleural cavity with regular X-ray examinations. However, these measures were ineffective. The patient was scheduled for surgery, and we intraoperatively observed an unusual cause of pneumothorax. Thus, we present spontaneous pneumothorax following right upper lobe pulmonary sequestration. The uniqueness of this case is associated with unusual manifestation and non-standard localization of rare lesion. A few cases of pneumothorax in similar patients are described in the world literature. The key limiting factor in diagnosis of such defects (identification of aberrant vessel supplying abnormal lung parenchyma) is the lack of routine CT angiography in patients diagnosed with pneumothorax. That is why CT changes were interpreted as postoperative ones, and the true cause was established only during redo surgery. A thorough inspection of the pleural cavity and alertness regarding unusual appearance of the right upper pulmonary lobe made it possible to suggest a non-standard diagnosis, avoid complications (bleeding from afferent vessel) and perform adequate lung resection.

Pulmonary sequestration in adult patients: a single-center retrospective study.

BACKGROUND: Pulmonary sequestration (PS) is a rare congenital lower airway malformation. This study presents the clinical and imaging features and surgical outcomes of PS in adults, and compare the safety and feasibility of minimally invasive surgery versus open thoracotomy for PS. METHODS: Adult patients with PS treated at our center from July 2011 to September 2021 were included. Information regarding the patient demographics, clinical and CT features, arterial supply and venous drainage, and surgical outcomes were collected. RESULTS: Ninety seven patients were included. The most common CT findings were mass lesions (50.5%) and cystic lesions (20.6%). The vast majority of the lesions (96 out of 97) were located close to the spine in the lower lobes (left vs. right: 3.6 vs. 1). Arterial supply was mainly provided by the thoracic aorta (87.4%) and abdominal aorta (10.5%). Intralobar and extralobar PS accounted for 90.7% and 9.3% of the patients, respectively. Three (4.5%) patients who underwent minimally invasive surgery were converted to open thoracotomy due to dense adhesions. Though no significant differences regarding operative time (P = 0.133), the minimally invasive surgery group was significantly better than the open thoracotomy group regarding intraoperative blood loss (P = 0.001), drainage volume (P = 0.004), postoperative hospital days (P = 0.017) and duration of chest drainage (P = 0.001). There were no cases of perioperative mortality. Only four (4.1%) patients developed postoperative complications, and no significant difference existed between the two groups. CONCLUSION: Our study revealed PS can present with a variety of different clinical and radiologic manifestations. Clinicians should consider the possibility of PS when diagnosing a lesion in the lower lobes close to the spine. Moreover, minimally invasive surgery is a safe and effective treatment modality for the treatment of PS in an experienced center.

Extralobar Pulmonary Sequestration in Adrenal Mimicking Neuroblastoma: A Case Report.

Background: Extralobar pulmonary sequestration (ELS) is a malformation composed of bronchopulmonary tissue outside the lungs that is discontinuous from the main tracheobronchial tree. ELS can present as a suprarenal space occupying lesion. Case Report: A 1-day old girl presented with a right supra-adrenal mass, first detected in-utero at 34 weeks. The differential included congenital neuroblastoma, but the urinary VMA was normal. At resection, there was lung tissue composed of dilated and tortuous bronchioles lined by columnar epithelium present in a back- to- back arrangement along with thick-walled vessels, features of an ELS with congenital pulmonary airway malformation type II. Conclusion: ELS enters the differential diagnosis of neonatal suprarenal masses with normal urinary catecholamines, and can have the morphology of a type II congenital pulmonary airway malformation.

[Intralobar Sequestration Associated with Aneurysm of the Aberrant Artery and Aspergillus Infection: Report of a Case].

A 32-year-old man was referred to our department for surgery because of recurrent pneumonia. Aspergillus fumigatus was detected in his sputum culture at the first visit. We started antifungal therapy one month before surgery. His chest radiograph showed an infiltrative shadow in the right lower lobe, and chest computed tomography (CT) showed an infiltrative shadow and large and small cystic changes in the right lower lobe. CT angiography (CTA) revealed two anomalous arteries supplied by the aorta that drained into the right lower lobe. An aneurysm with a diameter of 25 mm had formed in one anomalous artery. Based on these findings, intralobar pulmonary sequestration with Aspergillus infection and an anomalous artery forming an aneurysm was diagnosed. In addition, we embolized the aneurysm of the anomalous artery. After embolization, right lower lobectomy was safely performed. The patient was discharged on the 15th post-operative day with no complications.

Intrapulmonary sequestration with destructive pneumonia and life-threatening hemoptysis in an adult patient: a case report.

The article reports the case of a patient with bronchopulmonary sequestration complicated by destructive actinomycotic inflammation leading to life-threatening hemoptysis. It was an adult patient with the history of repeated right-sided pneumonia the cause of which had not been investigated in detail in the past. Only hemoptysis, which appeared as a complication, led to a closer investigation of the background of repeated right-sided pneumonia. CT scan of the chest revealed a lesion of the middle lobe of the right lung with anomalous vascularization - compatible with intralobar sequestration. Initially, conservative antibiotic treatment of pneumonia was provided at a local clinic. Embolization of the afferent vessels of the sequestrum was indicated due to persistent hemoptysis; this led to a reduction of its blood supply, proven by a follow-up CT examination of the chest. Clinically, the hemoptysis subsided. Three weeks later, the hemoptysis reocurred. The patient was acutely hospitalized at a specialized thoracic surgery department where shortly after admission, hemoptysis progressed to life-threatening hemoptea. Urgent middle lobectomy of the right lung was approached via thoracotomy to treat the source of bleeding. The case describes unrecognized bronchopulmonary sequestration as a possible cause of recurrent ipsilateral pneumonia in adulthood; additionally, it emphasizes the possible risks associated with a pathologically altered tissue microenvironment of pulmonary sequestration, and the need for surgical removal in all indicated cases.

Intralobular pulmonary sequestration in the middle lobe supplied by a right internal mammary artery: a case report.

BACKGROUND: Pulmonary sequestration (PS) is a rare congenital malformation that is more common in the left lower lobe, and the thoracic aorta is the most common arterial supply. CASE PRESENTATION: We describe a case of a 67-year-old man with a chief complaint of intermittent cough and hemoptysis who had been diagnosed by multidetector computed tomography angiography with right middle lobe intralobular pulmonary sequestration supplied by a right internal mammary artery. Finally, he underwent middle pulmonary lobectomy with normal postoperative recovery. DISCUSSION: This is a rare intralobular pulmonary sequestration case for a feeding artery from the right internal mammary. Multidetector computed tomography angiography should be performed for diagnosis and preoperative evaluation once pulmonary sequestration is suspected.

Debating the embolization of a large aberrant systemic artery for pulmonary sequestration using an Amplatzer duct occluder: a case report and literature review.

Here, we report two rare cases of pulmonary sequestration that were fed by large systemic arteries and embolized with a large Amplatzer duct occluder and their 3-year follow-up, and we discuss the efficacy and safety of the embolization of a large aberrant systemic artery to pulmonary sequestration using an Amplatzer duct occluder. A 9-year-old boy complained of chest pain for 1 month, and a 6-year-old boy initially complained of recurrent cough for 3 months. A series of examinations was launched to evaluate any possible malformation or abnormalities in the patients. Chest CT and CTA identified a right lower pulmonary sequestration with infection. After admission, transcatheter device occlusion was planned after essential antibiotic treatment, and postoperative infection prevention and anti-inflammatory treatment were given. In the following 2 years of follow-up, neither of the children had recurrent chest pain, cough or other related symptoms. However, the CT follow-up demonstrated that a residual mass was visible in both patients. The same chest scan section revealed slight reductions in lung lesions from 38.344 cm2 to 37.119 cm2 (3% reduction) and 14.243 cm2 to 13.178 cm2 (7.5% reduction) for each patient. No follow-up data demonstrated the long-term clinical outcomes of the residual lesion. We do not recommend that embolization be performed for large pulmonary sequestration lesions with an aberrant artery larger than 6 mm that is planned to receive a device larger than 10 mm, as their outcomes showed a higher possibility of rebuilding the vascularization network feeding the pulmonary sequestration, indicating a higher risk for long-term complications.

Comparing open and thoracoscopy approach for the treatment of pulmonary sequestration in children.

BACKGROUND: In this article, we are going to report our experience on thoracoscopy approach for pulmonary sequestration in children and compare it with traditional open approach. METHODS: This retrospective study included 25 patients with pulmonary sequestration (marked as Group 1), who were treated from January 2003 to December 2009 with open approach, and 32 patients treated between January 2010 and December 2019, who underwent thoracoscopy procedure (marked as Group 2). The preoperative data, operative details, and follow-up outcomes were studied and compared between two groups to discuss the advantages of thoracoscopy approach. Statistical comparison was made with unpaired t tests, χ2 tests and Fisher exact probabilities, with P value less than 0.05 considered significant. RESULTS: To bring less-related effect during the comparison, patients with similar preoperative data were selected. Group 2 whose operation time was longer than Group 1 was completed with thoracoscopy approach in all but one patient, who was converted to open surgery. However, the antibiotics consumption, drainage time, and hospital stay after surgery were shorter in Group 2 with less intraoperative blood loss, less postoperative complication and less volume of drainage. All patients were followed-up by CT scan for 3-24 months (mean, 13.8 months) and the recovery and improvement were better in Group 2. CONCLUSION: Compared to open surgery, thoracoscopy surgery has many benefits and was safe, feasible, and effective for the treatment of pulmonary sequestration in child patients.

Successful Treatment of Bronchopulmonary Sequestration by Radiofrequency Ablation of Feeding Artery.

INTRODUCTION: Most cases of bronchopulmonary sequestration (BPS) regress. Prenatal intervention is needed in cases of fetal hydrothorax or hydrops. Laser is commonly used to ablate the feeding artery. CASE PRESENTATION: In a fetus with BPS, radiofrequency ablation (RFA) was used to ablate the feeding artery arising from descending aorta at 29 weeks gestation. There was an extralobar BPS and significant pleural effusion causing mediastinal shift and collapse of lung. The effusion and tumor started decreasing from day 3 after procedure, and by the time patient delivered at 36 weeks gestation, the lesion had almost resolved. CONCLUSION: With proper technique, RFA can be safely used to ablate feeding artery in BPS.

[Congenital Malformations of the Lung - an Overview]. / Angeborene Fehlbildungen der Lunge ­ eine Übersicht.

Congenital pulmonary malformations comprise a heterogenous group of rare developmental diseases. The most common malformations are the tracheal bronchus, bronchial atresia, bronchogenic cyst, pulmonary sequestration, congenital lobar emphysema, and congenital pulmonary airway malformation. Due to their space-consuming effect, patients suffer early postnatal respiratory distress which generally requires immediate surgical resection. The management of asymptomatic lesions remains subject to debate, but early elective surgery is generally recommended to avoid respiratory and infectious complications at a later time point.We here provide a comprehensive review in which we present causes, clinical presentation and therapeutic options for the most prominent congenital malformations of the airways and lung parenchyma.

'Hybrid' Bronchopulmonary Malformation - Lobar Emphysema and Extra Lobar Sequestration.

Background: Idiopathic lobar emphysema (ILE) and bronchopulmonary sequestration (BPS) are two of the well-characterized pulmonary malformations. Case report: An antenatally detected case of a left bronchopulmonary malformation (BPM) was clinicoradiologically diagnosed to be a left upper lobar emphysema with isolated dextrocardia in the neonatal period. Besides the emphysematous left upper lobe, an accessory lobe akin to an extra lobar BPS was an operative surprise. Histopathological examination of both excised lobes led to a revised diagnosis of a 'hybrid' malformation comprising lobar emphysema and extra lobar BPS. The postoperative recovery was uneventful. Discussion/conclusion: The observations suggest that BPS and ILE may be interim entities in a continuum of abnormal embryogenesis. Such a hybrid malformation has not been hitherto reported.

A Modified VATS Procedure for Treating Adult Intralobar Pulmonary Sequestration.

Video-assisted thoracoscopic surgery (VATS) makes it possible to treat intralobar sequestration (ILS) more minimally invasive compared with conventional open surgery. However, this procedure is challenging to expose and isolate the aberrant arteries of ILS and the risk of bleeding is high. Herein, we developed a modified VATS procedure in which the aberrant vessels are treated in the last step of lobectomy, rather than at the beginning. In this way, we can expose the aberrant vessels easier and reduce the risk of massive blood loss, also simplifying the surgical procedure.

Antenatal Management of Bronchopulmonary Sequestration by Intrafetal Vascular Laser Ablation under Ultrasound Control: Narrative Review of the Literature and Report of Three Cases.

OBJECTIVE: The objective of this study is to assess the effectiveness and safety of intrafetal vascular laser ablation (VLA) for fetuses with bronchopulmonary sequestration (BPS) with hydrops. METHODS: First, we present 3 cases of fetuses with BPS and hydrops treated by VLA. Second, we aimed to conduct a narrative review to identify all reported cases of fetuses with BPS treated by intrafetal VLA. RESULTS: The review of the literature identified 41 fetuses treated by VLA for BPS with hydrops. The median gestational age of the VLA was 27+0 weeks' gestation [25+0-31+0] with an associated procedure at the same time in 43% of the cases (pleuroamniotic shunt, thoracentesis, and amniodrainage). A second procedure was required in 25% of cases for residual flow in the feeding vessel. No stillbirth or neonatal death was reported. The complications reported were a fetal thoracic hematoma complicated by fetal anemia and 4 preterm deliveries with a rate of 9%. CONCLUSION: VLA of the feeding vessel can be an effective treatment but is not without complications. In cases demonstrating cardiac output failure, intrafetal VLA should be considered as a treatment for BPS.

[Clinical analysis of pulmonary sequestration with torsion in 4 cases and literature review].

Objective: To summarize and analyze the clinical characteristics of children pulmonary sequestration with torsion. Methods: The main clinical manifestations, auxiliary examinations (chest B ultrasound, chest enhanced CT, pathological results), treatment and prognosis of 4 cases with pulmonary sequestration with torsion in Shenzhen Children's Hospital from November 2010 to November 2020 were retrospectively analyzed. Results: Among the 4 children, 2 were male and female,The age was 3 years and 8 months, 6 years and 9 months, 7 years and 5 months, 10 years old respectively.All the 4 cases were extralobar sequestrations, 2 cases were on the left side and 2 cases were on the right side. Abdominal pain was the first symptom, followed by chest pain in 2 cases, fever in 2 cases, shortness of breath in 1 case; 4 cases of chest color ultrasonography showed the soft tissue masses in the chest, medium volume pleural effusion in 2 cases, small pleural effusion in 2 cases, enhanced chest CT showed soft tissue mass without enhancement or slight enhancement in all 4 cases, and the blood supply artery was not clearly shown. Pathological results showed pulmonary sequestration with hemorrhage and necrosis, and 4 cases were cured after thoracoscopic surgery. Conclusions: Abdominal pain is often the first or main symptom of pulmonary sequestration with torsion in children, accompanied by or without chest pain. Chest ultrasound and CT can reveal the soft tissue density mass which is not enhanced or slightly enhanced during enhancement, and the blood supply artery is unclear. The effect of resection is well.

[Surgical Treatment for Congenital Bronchial Atresia and Intralobar Sequestration in the Same Lobe].

A 21-year-old man who had a history of pneumonia twice presented with chest discomfort. Computed tomography( CT) revealed dilatation of the atretic bronchus that was not continuous with the proximal one, and surrounding hyper-lucent lung fields in the outer and mediastinal sides of the right basal segment, and the absence of B10b+c. CT also demonstrated the presence of A10b+c in the former field and the aberrant artery from the inferior phrenic artery in the latter. Right basal segmentectomy was performed under the diagnosis of congenital bronchial atresia and intralobar sequestration. Pathological diagnosis accorded with preoperative one.

[VATS resection of basal pyramid segments in sequestration of the left lower lobe]. / Videotorakoskopicheskaya rezektsiya segmentov bazal'noi piramidy pri sekvestratsii nizhnei doli levogo legkogo.

Lung sequestration is a congenital malformation represented by a non-functioning pulmonary parenchyma supplied via an aberrant artery of systemic circulation. This malformation makes up 1-6% of all congenital lung abnormalities. In 1946, Pryce M.D. proposed the term «lung sequestration¼ for the disease first described by the Austrian pathologist C. Rokitansky in 1856. Lung sequestration becomes quire common due to development and availability of X-ray diagnostic methods. An aberrant artery can arise from the great vessels (thoracic and abdominal aorta) and smaller arteries (coronary, intercostal arteries). To date, there are 2 forms of lung sequestration (intralobar and extralobar). We report one of the options for surgical treatment of lung sequestration.

Video-Assisted Thoracoscopic Surgery for Pulmonary Sequestrations: Series of 35 Consecutive Patients in a Single Center.

OBJECTIVE: The aim of this report is to summarize the experience of completely video-assisted thoracoscopic surgery (VATS) for pulmonary sequestration in a single center and to evaluate the long-term outcome in a larger series of patients. METHODS: The data of 35 pulmonary sequestrations who received completely VATS consecutively in Peking University People's Hospital between January 2008 and November 2017 were retrospectively reviewed. Twenty-three females and twelve males with an average of 38 years old were included. RESULTS: A total of 28 (80%) patients had preoperative symptoms; leading symptoms were recurrent infections (22), fever (11), hemoptysis (11), chest pain (9), and shortness of breath (4). Twenty-nine (82.9%) patients were intralobar pulmonary sequestration (22 in the left lower lobe, 6 in the right lower lobe, and 1 in the left upper lobe) and six (17.1%) patients were extralobar pulmonary sequestration. All the patients underwent VATS excision successfully, 26 underwent lobectomy, 2 underwent wedge resection, 1 underwent occlusion of the aberrant artery, and 6 underwent mass resection in all of those with extralobar pulmonary sequestration. The median surgery time and estimated blood loss was 150 (75-300) minutes and 50 (10-600) mL, respectively. There was no mortality. Only one patient suffered postoperative complication (recurrent laryngeal nerve injury). During the median follow-up period of 57 months, none of the patients presented recurrence. CONCLUSIONS: Completely VATS was a safe and effective mini-invasive procedure for pulmonary sequestration in an experienced team. Its long-term outcome was remarkable.