Intralobar Pulmonary Sequestration Research Articles

Bilateral intralobar pulmonary sequestration in a newborn, case report and review of the literature on bilateral pulmonary sequestrations

We describe a rare case of bilateral intralobar pulmonary sequestration in a newborn with both sequestrations vascularized from a vessel arising from the celiac trunk of the abdominal aorta. The larger sequestration on the right side was diagnosed antenatally, the left side postnatally. At the age of 7 months, the child underwent successful bilateral thoracotomy. In follow-up at the age of more than 1 year, no complications have occurred.

Pulmonary sequestration: A single-institutional series composed of 27 cases

Large series about pulmonary sequestration from a single institute are rare in the literature. In this study, we aimed to evaluate diagnosis, treatment, and outcomes of pulmonary sequestration in a single institute. Records of patients with pulmonary sequestration between January 1982 and January 2006 were reviewed retrospectively. Age, sex, symptoms, diagnostic procedures, operative findings, operative techniques, postoperative complications, and follow-up results were evaluated. Twenty-seven patients, 17 male and 10 female, with an average age of 23.3 were operated on for pulmonary sequestration. Twenty patients had preoperative symptoms including recurrent pneumonia attacks, chest pain, hemoptysis, and shortness of breath. Chest radiography, thoracic computed tomography, aortography, magnetic resonance imaging, and bronchoscopy were used as diagnostic methods. Of the cases, 19 (70%) were intralobar pulmonary sequestration and 8 (30%) were extralobar pulmonary sequestration. Surgical procedures were lower lobectomy in 18 and segmentectomy in 1 of the patients with intralobar pulmonary sequestration and simple mass excision in all of those with extralobar pulmonary sequestration. Postoperative histopathologic examinations excluded any other alternative diagnosis. Furthermore, it detected an aspergilloma ball in 1 of the intralobar pulmonary sequestration specimens. Two patients had a postoperative complication (prolonged air leak in 1 patient and empyema in the other). During the follow-up period (mean 2.3 years), none of the patients presented a problem. No mortality was encountered. Owing to the potentially severe complications they can cause, pulmonary sequestrations should be removed whenever they are diagnosed. Since careful dissection provides sufficient surgical comfort, preoperative identification of the aberrant vessels is not a rule for the success of the operation.

An uncommon extrapulmonary sequestration located in the upper posterior mediastinum associated with the azygos lobe in a child

Pulmonary sequestrations are rare congenital anomalies with an incidence of 0.15% to 1.7%, and they are divided into intralobar and extralobar pulmonary sequestrations (ELPS). 1 A nonfunctioning pulmonary tissue characterizes them without normal connection to the tracheobronchial tree and supplying blood from 1 or more abnormal systemic arteries. Most ELPS are located between the lower lobe of the lung and the diaphragm; however, they may occur anywhere within the thorax and partially or completely within the diaphragm. 1,2 In approximately 25% of patients with ELPS, different congenital lung abnormalities, such as hypoplasia, congenital cystic adenomatoid malformation, congenital lobar emphysema, or bronchogenic cyst, associated with ELPS are known. 2 In this report, we present a child with an ELPS located in the upper posterior mediastinum associated with the azygos lobe.

INTRALOBAR PULMONARY SEQUESTRATION ASSOCIATED WITH A CONGENITAL PULMONARY AIRWAY MALFORMATION TYPE II

Intralobar pulmonary sequestration (ILPS) is a rare congenital malformation of the lung. It is described as a segment of lung parenchyma with normal pleura and systemic blood supply and it has poor communication with the tracheobronchial tree. Patients usually present in later childhood or adulthood with a history of recurrent pneumonias. The malformation is rarely associated with other congenital anomalies such as a congenital pulmonary airway malformation (CPAM). A CPAM is a congenital cystic lesion of the lung that presents usually in the newborn period as respiratory distress. We describe the case of a 2-month-old female who presented to the local emergency room with the initial diagnosis of pneumonia, surgical diagnosis of ILPS, and a final pathological diagnosis of ILPS with an associated CPAM.

低線量ラセンCTによる肺癌検診にて発見された肺葉内肺分画症の一例

A 52-year-old man underwent lung cancer screening with low-dose spiral computed tomography (CT) in a medical check-up at the Japanese Red Cross Kumamoto Health Care Center. He was asymptomatic. Chest x-ray on a medical check-up showed no abnormal shadows. CT scans revealed a nodule in the right lower lung, suggestive of its connection to the descending thoracic aorta. A diagnosis of pulmonary sequestration was considered. He was transferred to Kumamoto University Hospital for further examination. Contrast enhanced multidetector CT images demonstrated that a nodule in the right lower lobe and an anomalous artery ran from the descending thoracic aorta, flowed through the right lower lobe and returned to the right inferior pulmonary vein. Intralobar pulmonary sequestration was confirmed by contrast enhanced multidetector CT. We report this case of asymptomatic intralobar pulmonary sequestration diagnosed using contrast enhanced multidetector CT.

下横隔動脈より血液供給を受けた肺葉内肺分画症と思われる１例

下横隔動脈より血液供給を受けた肺葉内肺分画症と思われる１例

７９歳で発見された気管支原性嚢胞を合併した肺葉内肺分画症の１切除例

症例は79歳，男性．健診で右肺野の異常陰影を指摘され入院した．胸部CTおよびMRIでは，上縦隔に嚢胞性腫瘤影と右肺下葉に左胃動脈から分岐する異常血管が流入する腫瘤影が認められた．動脈造影検査でも左胃動脈から分岐する異常動脈を確認した．以上よりPryce III型肺葉内肺分画症および上縦隔嚢胞と診断し，手術を施行した．胸腔鏡補助下に異常動脈の切離，分画肺の切除と上縦隔の嚢胞摘出を行った．本症例は異常動脈が左胃動脈から起始し，さらに縦隔気管支原性嚢胞を合併した極めて稀な症例と考えられた．両疾患が合併している点から考察すると，肺葉内肺分画症は胎生前期に前腸から異常発芽した副肺芽に起因し，二次的に体循環からの動脈血流を受けた先天性疾患である可能性もあると考えられた．

3D multidetector CT angiographic evaluation of extralobar pulmonary sequestration with anomalous venous drainage into the left internal mammary vein in a paediatric patient

Pulmonary sequestration is a congenital lung malformation, defined by dysplastic and non-functioning lung tissue lacking normal tracheobronchial connections and accompanied by an anomalous systemic blood supply. Recognition of anomalous arteries and veins in pulmonary sequestration is paramount to making the correct diagnosis. In contrast to intralobar pulmonary sequestration, where anomalous venous drainage is usually into the pulmonary venous system, the pattern of anomalous venous drainage is more varied in extralobar pulmonary sequestration. To the best of our knowledge, anomalous venous drainage to the internal mammary vein in extralobar sequestrations has not been reported. We report an anomalous venous drainage into the internal mammary vein in an extralobar sequestration which was evaluated with 3D multidetector CT angiography.

Video-assisted resection of bilateral intralobar pulmonary sequestrations

Bronchopulmonary sequestrations (BPSs) are characterized by anomalous vascular and tracheobronchial connections and account for a small proportion of pulmonary malformations. Bilateral BPSs are extremely rare, and the traditional surgical approach has been sequential resections with staged thoracotomies. We report a single-stage, sequential, video-assisted thoracoscopic surgery (VATS) approach to bilateral intralobar pulmonary sequestrations (ILSs). An otherwise healthy 7-year-old girl was referred with low-grade fevers, productive cough, and a right lower lobe infiltrate on standard chest radiographs. She was given 2 courses of antibiotics, with only partial resolution of her right lung infiltrate. Subsequent high-resolution computed tomographic imaging demonstrated bilateral lower lobe BPS (Figure 1, A). Arterial supply to both sides was from the abdominal aorta (Figure 1, B). In the operating room, after achievement of general anesthesia with a single-lumen endotracheal tube and bronchial blocker, the patient was initially placed in the left lateral decubitus position. The lung was carefully inspected with a 5-mm, 30° thoracoscope through a 5-mm anterior ninth interspace thoracoport. A 5-cm access incision was made in the fifth interspace anteriorly, sparing the serratus anterior muscle. Visualization was improved by placing a figure-of-eight stitch in the tendinous portion of the diaphragm and drawing it down through the same ninth interspace thoracoport. A consolidated intralobar sequestration was noted in the posterior and lateral basilar segments of the right lower lobe, with the aberrant arterial supply located in the inferior pulmonary ligament. The arterial supply was dissected and divided with a vascular TA-30 stapler. A sequestrectomy was performed with multiple applications of an Endo GIA 3.5-mm linear stapler (Figure 2). A 20F chest tube was placed, the lung was inflated under direct vision, and the incisions were closed in layers. The patient was then turned to the right lateral decubitus position, access to the left pleural space was obtained through a 5-mm ninth interspace thoracoport, and a 5-cm access incision was made in the fifth interspace anteriorly. The lung was inspected, and a similar intralobar sequestration was observed in the posterior basilar segment of the left lower lobe. The aberrant arterial source in the inferior pulmonary ligament was isolated and divided, and sequestrectomy was performed in similar fashion. A single left chest tube was placed, and the patient was transferred to the pediatric intensive care unit. Both chest tubes were removed on the second postoperative day, and the patient was discharged to home on the fourth postoperative day. Final pathology showed bilateral intralobar sequestrations with severe inflammation. ILSs are characterized by nonfunctioning lung tissue that lacks normal communication with the tracheobronchial tree. The arterial supply to ILSs is most often from the abdominal aorta, and venous drainage is usually through normal routes to the left atrium. Although children with BPSs are at risk for associated abnormalities, they occur less with intralobar than with extralobar sequestrations. To date, there have been 12 reported cases of bilateral BPS in the literature, with only 5 intralobar cases.1Spinella P.C. Strieper M.J. Callahan C.W. Congestive heart failure in a neonate secondary to bilateral intralobar and extralobar pulmonary sequestrations.Pediatrics. 1998; 101: 120-124Crossref PubMed Scopus (10) Google Scholar All operative cases were approached with staged sequential thoracotomies.1Spinella P.C. Strieper M.J. Callahan C.W. Congestive heart failure in a neonate secondary to bilateral intralobar and extralobar pulmonary sequestrations.Pediatrics. 1998; 101: 120-124Crossref PubMed Scopus (10) Google Scholar More recently, surgical resection of BPSs has been accomplished with a VATS approach.2Wan I.Y. Lee T.W. Sihoe A.D. Ng C.S. Yim A.P. Video-assisted thoracic surgery lobectomy for pulmonary sequestration.Ann Thorac Surg. 2002; 73: 639-640Abstract Full Text Full Text PDF PubMed Scopus (39) Google Scholar, 3Sakuma T. Sugita M. Sagawa M. Ishigaki M. Toga H. Video assisted thoracoscopic wedge resection for pulmonary sequestration.Ann Thorac Surg. 2004; 78: 1844-1845Abstract Full Text Full Text PDF PubMed Scopus (27) Google Scholar, 4Klena J.W. Danek S.J. Bostwick T.K. Romero M. Johnson J.A. Video-assisted thoracoscopic resection for intralobar pulmonary sequestration single modality treatment with video-assisted thoracic surgery.J Thorac Cardiovasc Surg. 2003; 126: 857-859Abstract Full Text Full Text PDF PubMed Scopus (15) Google Scholar Lobectomy, sequestrectomy, and wedge resections have all been described, with excellent results and a small number of conversions to thoracotomy.5de Lagausie P. Bonnard A. Berrebi D. Petit P. Dorgeret S. Guys J.M. Video-assisted thoracoscopic surgery for pulmonary sequestration in children.Ann Thorac Surg. 2005; 80: 1266-1269Abstract Full Text Full Text PDF PubMed Scopus (39) Google Scholar Because disease in this patient was limited to the basilar segments of the lower lobes, it was possible to perform sequestrectomies and preserve lung parenchyma. This unique case of bilateral ILS demonstrates the feasibility of a single-setting, sequential VATS approach to minimize postoperative pain and limit multiple or prolonged hospitalizations.

Preoperative magnetic resonance imaging for intralobar pulmonary sequestration

Preoperative magnetic resonance imaging for intralobar pulmonary sequestration

Pulmonary sequestration with high levels of tumor markers tending to be misdiagnosed as lung cancer

A 62-year-old man with hemoptysis and an abnormal shadow on chest roentgenogram was diagnosed as having anomalous systemic arterial supply to the normal basal segment of the left lower lobe. The preoperative serum carbohydrate antigen 19-9 and carcinoembryonic antigen levels were 73.8 units/ml and 10.8 ng/ml, respectively. Histopathological examination confirmed that the lesion was an intralobar pulmonary sequestration without air connection. There was no malignant finding in the resected specimen. The serum values of tumor markers returned to their approximate normal ranges after lower lobectomy.

Intralobar pulmonary sequestration receiving arterial supply from the superior mesenteric artery: A case report

We report a case of congenital anomaly of pulmonary sequestration by contrast-enhanced three-dimensional magnetic resonance (MR) angiography and digital subtraction angiography (DSA). The pulmonary sequestration receives a blood supply from the superior mesenteric artery. MR angiography technique has been employed in pulmonary sequestration.

1 Case of Bronchial Leiomyoma with Intralobar Pulmonary Sequestration

Both bronchial leiomyoma and pulmonary sequestration are rare conditions, and to the best of our knowledge there are no reports of the two conditions coexisting. We report a female patient with bronchial leiomyoma with acquired pulmonary sequestration who presented with dyspnea, cough and purulent sputum. The patient had been treated for pneumonia at a local medical clinic. but was transferred to our clinic beacausr there was no clinical improvement. A 3-D computed tomography scan revealed a 1.5 cm sized mass near the distal portion of the left main bronchus and an anomalous artery arising from the aorta. The patient showed clinical improvement after a left lower lobectomy and a ligation of the anomalous artery.

A Case of Right Upper Lobe Intralobar Pulmonary Sequestration Associated with Right Tracheal Bronchus

A Case of Right Upper Lobe Intralobar Pulmonary Sequestration Associated with Right Tracheal Bronchus

MDCT Demonstration of Intralobar Pulmonary Sequestration of the Right Upper Lobe in an Adult

MDCT Demonstration of Intralobar Pulmonary Sequestration of the Right Upper Lobe in an Adult

Intralobar Pulmonary Sequestration With Arterial Supply from the Coronary Circulation

Pulmonary sequestration is an unusual malformation. It consists of a nonfunctioning mass of lung tissue that shows no normal continuity with the tracheobronchial tree and derives its arterial blood supply from the systemic circulation. The thoracic and abdominal aortas are the most common sites of origin of the abnormal nutrient branches. Arterial supply of a sequestration from the coronary circulation is extremely rare. We present a case of a right middle lobe sequestration deriving its branches from the left coronary artery.

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.

Intralobar pulmonary sequestration diagnosed by MR angiography

A definitive diagnosis of pulmonary sequestration necessitates evidence of a systemic arterial supply to the sequestered lung segment by aortography. We report a case of intralobar pulmonary sequestration in an 11-month-old girl in whom the diagnosis was made by contrast-enhanced magnetic resonance (MR) angiography. Contrast-enhanced MR angiography is the most reliable noninvasive method for the definitive diagnosis of pulmonary sequestration in children.

Seqüestro broncopulmonar intralobar: relato de caso

Neste trabalho é descrito um caso de uma paciente de 49 anos de idade que apresentava infecções pulmonares de repetição. Na telerradiografia de tórax observou-se massa no segmento basal posterior do pulmão direito, que na angiotomografia e nas reconstruções em 3D notou-se a presença de um suprimento sanguíneo proveniente da artéria aorta descendente. No exame da peça cirúrgica ratificou-se a presença de seqüestro pulmonar intralobar com cavidade preenchida por muco.

Pulmonary sequestration and aspergillosis

Pulmonary sequestration (PS) is an uncommon congenital disease. Symptoms when present are usually secondary to pyogenic infection. Our objective was to draw attention on superimposed fungal infection. During the last 20 years, we operated upon 19 intralobar PS. Four of six patients operated during the last decade proved to have intralobar PS containing Aspergillus. In one patient aspergillosis presented as an aspergilloma and communications between the sequestration and small bronchi were present. In another patient diagnosis was made before operation because of positive precipitins test. In two patients aspegillosis was discovered by the pathologist. Review of literature demonstrates PS fungal colonization to be a rare but recently reported entity (14 cases reported). The observation of four consecutive patients leads us to suggest that PS aspergillosis must be considered in order to evaluate its incidence.