Pulmonary Sequestration In Adult Research Articles

The diagnosis and surgical management of pulmonary sequestration in adults: a case series from a single centre in the UK

Pulmonary sequestration (PS) is a rare congenital malformation where extrapulmonary lung tissue receives systemic blood supply from an anomalous branch directly from the thoracic or abdominal aorta. Whilst non-malignant, it can often present with similar symptoms as lung cancer. We present a retrospective review of 8 consecutive adult patients undergoing surgical management for PS within a single centre in the UK. Of our cohort, 62.5% had never smoked. PS in the right lung was seen in 62.5% of cases. Anomalous branches of the pulmonary artery, pulmonary vein or coeliac axis supplied 37.5% of the PS seen in our cohort, and 12.5% did not have a radiologically identifiable blood supply. Techniques varied from thoracotomy (n = 4), video-assisted thoracoscopic surgery (VATS) (n = 3) to robotic resection (n = 1) with no intra-operative or post-operative complications reported within hospital. The mean length of stay was 2 days. The post-operative mortality rate was 12.5%; one patient had died following the robotic resection of the mass of pneumonia in the local district hospital 26 days post-operatively after being discharged. No other complications nor recurrence was recorded over the follow-up period. Where pulmonary masses receive blood supply from anomalous branches of the pulmonary vein and coeliac axis, diagnoses of PS should be considered. The clinical feasibility of discharge in 2 days with no symptom recurrence should undergo further investigation with a larger sample size.

Comparison of clinical outcomes of pulmonary sequestration in adults between surgery and non-surgery groups.

Pulmonary sequestration (PS) is a rare congenital lung malformation that can be incidentally diagnosed in adulthood. The natural course of PS in adults is scarcely known. In this retrospective cohort study, medical records and imaging results of adult patients diagnosed with PS between 1994 and 2019 were reviewed. Diagnoses of PS were confirmed by histopathological findings in resected cases, while non-resected cases were diagnosed based on the presence of anomalous systemic arterial supply and abnormal lung parenchyma on enhanced chest computed tomography (CT). Among 104 patients with PS, the median age at diagnosis was 40.5 years, and 69 (66.3%) patients were asymptomatic. Patients in the surgery group were significantly younger (38.6 vs. 45.3 years, respectively, P=0.016), were more likely to be symptomatic initially (51.6% vs. 28.6%, respectively, P=0.015), and had larger PS (90.0 vs. 66.3 mm, respectively, P<0.001) than the non-surgery group. Of the patients in the surgery group, 29.0% (18/62) experienced postoperative complications. In the surgically resected cases, infections were only detected in intralobar PS, not in extralobar PS. Among 25 subjects without initial symptoms in the non-surgery group, 24 (96.0%) remained asymptomatic at the last follow-up. Adults with PS tended to undergo resection if they were young, symptomatic, and had large PS (a median diameter of 90.0 mm). Almost all subjects who were initially asymptomatic and did not undergo surgery remained asymptomatic at the last follow-up. Therefore, considering the indolent course of PS, initially asymptomatic adults with PS could be followed up without surgery.

Indocyanine Green to Visualize and Resect Pulmonary Sequestration in Adults

Pulmonary sequestration (PS) is a rare congenital pulmonary dysplasia which presents with recurrent chest infections and haemoptysis. Its diagnosis is difficult and suspected on contrast-enhanced chest CT. The definitive treatment of pulmonary sequestration is surgical resection or endoluminal occlusion of the abnormal feeding vessel. Here, we report an innovative technique in which per-operative indocyanine green guided excision of intra-lobar sequestrated lung parenchyma and ligation of abnormal feeding vessel. The first patient was a 44-year-old gentleman with recurrent bouts of cough for 2 years. Courses of antibiotics failed to resolve it. Fibreoptic bronchoscopy and routine blood tests did not show any abnormality. CECT chest showed multiloculated cystic solid and low density mass in the right lower lung. The area of the lesion received its blood supply from anomalous systemic artery originating from coeliac trunk. Intraoperative, indocyanine green was injected into the atypical vessel, and pulmonary sequestration turned green. A sequential atypical lung resection could be completely removed with parenchyma preservation. The second patient was a 53-year-old lady presenting with chest pain, cough and expectoration for 2 years not responding to general management and antibiotics. CECT showed a low-density mass in the right lower lung lobe, with a feeding arterial vessel arising from the abdominal aorta. This was diagnosed as PS and resected using per-operative indocyanine green with parenchymal preservation and ligation of feeding vessel. Both patients have been followed up for at least 1 year and are symptom free. The intraoperative use of indocyanine green could greatly facilitate the presentation of the extent of a lung sequestration and safe resection.

Patent Ductus Arteriosus Can Coexist With Pulmonary Sequestration in Adults, Conditioning Pulmonary Hypertension

A woman in her early 40s attended the hospital for dyspnea. Her chest radiograph revealed cardiomegaly, prominence of the cone of the pulmonary artery, and hyperflow (Figure A). The electrocardiogram revealed growth of the atrium and right ventricle. The chest computed tomography angiogram depicted right intralobar pulmonary sequestration (PS) with a patent ductus arteriosus (Figure B); the blood supply of the sequestrum originated from the abdominal aorta with venous drainage toward the pulmonary vein and the left atrium (Figure C and D).

Endovascular embolization for pulmonary sequestration in adults: An adjunctive technique to delay surgical intervention

Pulmonary sequestration is a congenital malformation characterized by a non-functioning segment of dysplastic lung that lacks communication with the bronchial tree and contains an aberrant systemic arterial supply. Pulmonary sequestration is classically managed surgically; however, surgical intervention is not always feasible on an emergent basis. We present two cases, in support of recent literature, that reinforce the use of transcatheter embolization to achieve hemostasis and delay surgical intervention.

Surgical treatment of pulmonary sequestration in adults and children: long-term results.

Few experiences comparing paediatric and adult patients treated for pulmonary sequestration (PS) have been reported. Surgical treatment is considered the best choice, but the time of surgery is still controversial. We present our experience in this setting, comparing characteristics, histological results and outcome of paediatric and adult patients undergoing PS resection. Between 1998 and 2017, a total of 74 patients underwent lobectomy or sublobar resection for PS. Sixty patients were children (group A: ≤16 years old) and 14 were adults (group B: >16 years old). Preoperative diagnosis was radiological. PS was intralobar (42 cases) and extralobar (32 cases). The operation was a muscle-sparing lateral thoracotomy or video-assisted thoracoscopic surgery. Preoperative characteristics, histological results and short-/long-term results of the 2 groups were retrospectively analysed and compared. Thirty-seven percent of the patients in group A presented with respiratory symptoms and 79% in group B (P = 0.44). Most symptomatic patients were treated with a lobectomy. In group A, 2 patients (3%) had a malignant transformation of the lesion. Patients with a prenatal diagnosis treated after the age of 1 year became more symptomatic than those operated on before the age of 1 year (57% vs 23%; P = 0.08). No differences were found in postoperative complications. Long-term stable remission of respiratory symptoms was obtained in 91% of patients in group A and 100% in group B. Adulthood (P = 0.03) and the association with congenital cystic adenomatoid malformation (P = 0.03) were negative prognostic factors for the development of respiratory symptoms. Surgical treatment of PS is safe and feasible. Despite the small number of patients included, study results indicated that an early operation during childhood may prevent the subsequent development of respiratory symptoms. Surgical treatment is also recommended to prevent the rare transformation into malignancy.

INTRAPULMONARY SEQUESTRATION IN AN ADULT

SESSION TITLE: Tuesday Fellows Case Report Posters SESSION TYPE: Fellow Case Report Posters PRESENTED ON: 10/22/2019 01:00 PM - 02:00 PM INTRODUCTION: An intrapulmonary sequestration is defined as nonfunctioning lung tissue that is not in normal continuity with the tracheobronchial tree and derives its blood supply from systemic vessels[1]. Pulmonary sequestration is considered a childhood disease; however, the condition can continue into adolescence and sometimes adulthood[2]. Here, we present a middle-aged female presenting with pneumonia, found to have intrapulmonary sequestration and underwent lobectomy. CASE PRESENTATION: 43-year-old female with past surgical history of diaphragmatic hernia repair in childhood presented with persistent cough of two weeks duration with clear rhinorrhea and sharp left lower chest pain. Chest radiography revealed a left lower lobe consolidation suspicious for pneumonia. Patient was admitted for suspected pneumonia, and initial vital signs showed temperature 99F, heart rate 134, respiratory rate 25, blood pressure 97/75, and oxygen saturation of 99% on room air. Laboratory studies were significant for WBC count of 9.60, Hgb 10.5, and platelet count 69,000, with a d-dimer of 722. A CT angiogram to rule out a pulmonary embolism displayed a complete collapse of the left lower lobe with abnormal systemic arterial branch extending to the left lower lobe consistent with sequestration. Subsequently, patient underwent bronchoscopy that was noted to be normal with negative cultures and washings. The patient signed out against medical advice after the procedure, but returned to the hospital five days later with non-resolution of her cough and left sided chest pain. Chest radiograph and CT angiography performed showed unchanged findings of left lower lung infiltrate and suspected sequestration. Cardiothoracic surgery was consulted and patient was taken for a left lower lobectomy. The procedure and pathology confirmed the diagnosis of intrapulmonary sequestration. The patients’ symptoms resolved after surgery and she was discharged home in stable condition. DISCUSSION: Pulmonary sequestration, a disease of childhood, most commonly presents as pneumonia but may also present with chest pain, cough, shortness of breath and hemoptysis [3,4]. The condition may also be found incidentally in asymptomatic adults. Most pulmonary sequestrations arise in the left lung with two-thirds arising in the lower left lung lobe with the blood supply coming from systemic circulation, usually the thoracic or abdominal aorta. The treatment for a pulmonary sequestration involves surgical resection of the effected lung lobe to avoid infection and damage to the lung parenchyma. Our case is made unique by the advanced age of our patient with no prior history of recurrent infections or underlying respiratory symptoms prior to this presentation[5]. CONCLUSIONS: Pulmonary sequestration should be in the differential even in advanced age without prior history of underlying respiratory symptoms or recurrent infections. Reference #1: 1. "Congenital Malformations and Genetic Disorders of the Respiratory Tract.” American Review of Respiratory Disease, 120(1), pp. 151–185 Reference #2: 2. Petty, L., Joseph, A., & Sanchez, J. (2018). Case report: Pulmonary sequestration in an adult. Radiology Case Reports,13(1), 21-23. https://doi.org/10.1016/j.radcr.2017.09.029 Reference #3: 3. Frazier, A. A., Christenson, M. L., Stocker, J. T., & Templeton, P. A. (1997). Intralobar sequestration: Radiologic-pathologic correlation. RadioGraphics, 17(3), 725-745. https://doi.org/10.1148/radiographics.17.3.9153708 4. Hertzenberg, Casey & Daon, Emmanuel & Kramer, Jeffrey. (2012). Intralobar pulmonary sequestration in adults: Three case reports. Journal of thoracic disease. 4. 516-9. 10.3978/j.issn.2072-1439.2012.06.07. 5. Alsumrain, M., & Ryu, J. H. (2018). Pulmonary sequestration in adults: A retrospective review of resected and unresected cases. BMC Pulmonary Medicine, 18(1) DISCLOSURES: No relevant relationships by Anas Al-khateeb, source=Web Response No relevant relationships by Sharath Bellary, source=Web Response No relevant relationships by Michael Ellis, source=Web Response No relevant relationships by Muqueet Kadri, source=Web Response No relevant relationships by William Meng, source=Web Response No relevant relationships by Richard Miller, source=Web Response No relevant relationships by Rutwik Patel, source=Web Response No relevant relationships by Hari Sharma, source=Web Response No relevant relationships by Euclid St.Hill, source=Web Response

Pulmonary sequestration in adults: a retrospective review of resected and unresected cases

BackgroundPulmonary sequestration (PS) is a form of congenital pulmonary malformation that is generally diagnosed in childhood or adolescence and usually resected when diagnosed. We aim to identify the clinical presentation and course of patients diagnosed to have PS during adulthood.MethodsUsing a computer-assisted search of Mayo clinic medical records, we identified adult patients with PS diagnosed between 1997 and 2016. Clinical and radiological data were collected including postoperative course for those who underwent surgical resection.ResultsWe identified 32 adult patients with PS; median age at diagnosis was 42 years (IQR 28–53); 17 patients (53%) were men. The median sequestration size was 6.6 cm (IQR 4.4–9.3). The type of sequestration was intralobar in 81% and extralobar in 19%. The most common location was left lower lobe posteromedially (56%). Forty-seven percent of the patients presented with no relevant symptoms. The most common radiographic finding was mass/consolidation in 61% and the most common feeding artery origin was the thoracic aorta (54%). Surgical resection was performed in 18 patients (56%) and postoperative complication was reported in 5 patients (28%). There was no surgical mortality. Median duration of follow-up after diagnosis for unresected cases, most of whom were asymptomatic, was 19 months (IQR 4–26) with no complications related to the PS reported.ConclusionsNearly one-half of adult patients with PS present with no relevant symptoms. The decision regarding surgical resection needs to weigh various factors including clinical manifestations related to PS, risk of surgical complications, comorbidities, and individual patient preferences.

Pulmonary Sequestration in Adults: A Retrospective Review of Resected and Unresected Cases at a Tertiary Care Center

Pulmonary Sequestration in Adults: A Retrospective Review of Resected and Unresected Cases at a Tertiary Care Center

Extralobar pulmonary sequestration in adults

Pulmonary sequestration is defined as a nonfunctioning mass composed of dysplastic lung parenchyma, embryologically detached from the tracheobronchial tree and receiving its own blood supply from a systemic artery, usually the thoracic or abdominal aorta. It may be intralobar or extralobar depending on the presence of an independent pleural envelope. Extralobar sequestration is a much rarer entity, and diagnosis is usually made antenatally or in early life. Here, we present a rare case of extralobar pulmonary sequestration in an adult.

Pulmonary Sequestration: A Congenital Abnormality Seen in the Adult Pulmonologist’s Office

SESSION TITLE: Fellow Case Report Poster - Genetic and Developmental Disorders SESSION TYPE: Affiliate Case Report Poster PRESENTED ON: Tuesday, October 25, 2016 at 01:30 PM - 02:30 PM INTRODUCTION: Pulmonary sequestration is a rare congenital malformation. Diagnosis is often delayed until the 2nd or 3rd decade. Common symptoms including recurrent infections and hemoptysis. Surgical resection is usually recommended due to the risk of massive hemoptysis. The following is a case of a 42 year old male presenting with his sentinel event of sequestration, which underscores the importance of adult pulmonologists recognizing congenital conditions. CASE PRESENTATION: A 42 year old male, with minimal past medical history, presented with small volume hemoptysis. In the Emergency Department, he was hemodynamically stable without hypoxia. CT of the chest with IV contrast was obtained due to the concern of pulmonary embolus. The CT showed ground glass infiltrates in the medial right lower lobe with an aberrant feeding vessel arising from the thoracic aorta. He was discharged in stable condition with pulmonary follow up. DISCUSSION: Pulmonary sequestration is a rare finding accounting for about only 5% of congenital pulmonary malformations. It is characterized by dysplastic lung tissue fed by an aberrant artery arising directly from the aorta. In extralobar sequestration, there is a distinct visceral pleura surrounding the dysplastic parenchyma. It is usually diagnosed in vitro, but often does not cause symptoms until adulthood. On imaging, it is commonly seen as a mass because it is rarely aerated. Intralobar sequestration, on the other hand, is more common and is not surrounded by an independent visceral pleura. It is more likely to result in recurrent infection, and therefore, earlier presentation in childhood. Intralobar sequestration imaging shows aerated but abnormal lung tissue. Both forms of sequestration usually require surgical resection due to the risk of massive, life-threatening hemoptysis. Standard thoracotomy or video assisted thoracic surgery (VATS) can be performed. There is limited data but increasing interest in endovascular intervention in adults. CONCLUSIONS: Pulmonary sequestration is a congenital malformation, but often presents in the adult pulmonologist’s office. Recognition is imperative because intervention can be life-saving. Reference #1: Qian X, Sun Y, Liu D, Wu X, Wang Z, and Tang Y. 2015. Pulmonary sequestration: a case report and literature review. Int J Clin Exp Med. 8(11):21822-21825. Reference #2: Leonici G, Rossi UG, Ferro C, and Chessa L. 2011. Endovascular treatment of pulmonary sequestration in adults using Amplatzer® vascular plugs. Interact Cardiovasc Thorac Surg. 12:98-100. DISCLOSURE: The following authors have nothing to disclose: Laurie Manka No Product/Research Disclosure Information

Intralobar pulmonary sequestration in adults: three case reports.

Pulmonary sequestration is a relatively rare entity comprising a small portion of all congenital pulmonary malformations. Varying clinical techniques have been utilized to manage this disease process including abstaining from surgical intervention, endovascular procedures and operative approaches. We reviewed three case reports with varying presentations, each of which had a favorable outcome with definitive surgical treatment. In conclusion, we recommend that resection should continue to be the standard of care in both adolescent and adult patients with this disease process.

Extralobar Pulmonary Sequestration With Hemorrhagic Infarction in an Adult

Symptomatic extralobar pulmonary sequestration in adults is thought to be extremely rare. We present a case of an extralobar pulmonary sequestration, with infarct, in a 28-year-old woman who complained abrupt onset of chest pain. Chest computed tomography and magnetic resonance imaging showed a mass in the left paraspinal area, medial to the left lower lobe, and serial chest radiographs revealed increasing pleural effusion. This was histopathologically confirmed to be a hemorrhagic infarct of the extralobar pulmonary sequestration.

Intraabdominal Pulmonary Sequestration

Bronchopulmonary sequestration is an uncommon developmental abnormality that frequently presents as an incidental mass. Differential diagnosis includes malignancy. Reports of extralobar abdominal pulmonary sequestration in adults are sparse. Only 2.5% of all pulmonary sequestrations are detected below the diaphragm. The authors present a case of a 45-year-old woman and a literature review. Computed tomography, magnetic resonance imaging, and operative and pathologic findings are provided. A review of diagnosis and therapy is presented.