Coil embolization to treat pulmonary sequestration in the right upper lobe.

Although there have been a few case reports of pulmonary sequestration, it is primarily located in the lower lobe and left lung, rarely in the right upper lobe. Here, we report a case presented with haemoptysis. Computed tomography images revealed flake ground-glass shadows in the right upper lobe. Computed tomography angiography demonstrated an artery supplied the affected lesions stemmed from the aortic arch. We diagnosed and treated her with bronchial artery angiography with coil embolization. No complications were found after operation until now. Thus, CTA could help identify the abnormal blood vessels, and interventional therapy may be an effective alternative to surgery of pulmonary sequestration.

Clinical Outcomes of Transarterial Embolization in the Treatment of Pulmonary Sequestration.

PURPOSE: To assess the safety and efficacy of transarterial embolization (TAE) for pulmonary sequestration (PS). METHODS: A single-center, retrospective study was conducted from March 2013 and December 2020. Patient characteristics, laboratory/imaging, complications, and the TAE procedure were reviewed. RESULTS: We report 11 symptomatic patients with PS successfully treated by TAE. The aberrant supplying arteries were embolized, and there were no immediate complications. One to three days after TAE, patients complained of mild chest pain (n = 4), moderate chest pain (n = 3), and low-grade fever (n = 1). Chest pain symptoms were completely resolved after 2-4 days. One patient with PS and bronchiectasis required thoracoscopic resection due to continued symptoms. The remaining 10 patients showed disappearance of chest pain and hemoptysis and decreased lesion size at 2 weeks and 3 months, with a clinical success rate of 90.9%. CONCLUSIONS: TAE may be a feasible alternative treatment for pulmonary sequestration.

Combined endovascular and surgical approaches to treat intralobar pulmonary sequestration: a case report.

Pulmonary sequestration is a congenital abnormality of a non-functional pulmonary mass with anomalous systemic arterial supply. Surgical resection is the gold standard treatment, but it carries a risk of life-threatening haemorrhage from accidental injury of the anomalous artery. Endovascular embolisation has been introduced as a safe alternative, but does not eliminate the possibility of symptom recurrence. We report a case of a 61-year old woman with intralobar pulmonary sequestration treated with a combination of endovascular coil embolisation and surgical resection.

Hybrid and Endovascular Treatment of Pulmonary Sequestration: Two Case Reports and Literature Review.

Pulmonary sequestration is an uncommon congenital pulmonary anomaly associated with aberrant systemic arteries which usually originate from the thoracic aorta or abdominal aorta. Traditionally, surgical resection and ligation of the feeding vessels are the gold standard treatments of the disease. Endovascular intervention and hybrid operation are promising treatment options. However, the case reports with endovascular and hybrid treatment are sparse to our knowledge. We presented 2 symptomatic adult patients with pulmonary sequestration successfully treated by hybrid operation and transcatheter embolization, respectively. Besides, we reviewed 37 previously reported cases of pulmonary sequestration treated by endovascular or hybrid treatment.

Non-operative management of extralobar pulmonary sequestration: a safe alternative to resection?

PURPOSE: This retrospective cohort study compares the natural history of patients with extralobar sequestrations (ELS) who do not undergo intervention with those who undergo resection to assess the safety of non-operative management. METHODS: 126 patients with pulmonary sequestrations or congenital pulmonary airway malformations born between 1999 and 2016 were identified. 49 patients had ELS on postnatal imaging, but two were excluded for associated congenital diaphragmatic hernia. Demographic and clinical data were retrospectively reviewed, with phone follow-up for non-operative patients with no records for > 1 year. Statistical analysis was by Fisher's exact test or Wilcoxon signed-rank test (two-tailed p < 0.05). RESULTS: 40% (19/47) were managed non-operatively and 60% (28/47) underwent resection. Non-operative patients were less likely to have an intrathoracic ELS: 47% (9/19) vs. 75% (21/28), p = 0.07. No symptoms were attributable directly to the ELS. Non-operative patients had median follow-up 3.2 years, during which time 88% (15/17) of ELS decreased in size on serial imaging. For patients who underwent resection, there was 100% concordance between imaging and intraoperative findings. There was no evidence of inflammation, infection or malignancy on final pathology, though 57% (16/28) of resected lesions had foci of non-aerated cysts. CONCLUSIONS: Although further longitudinal study is required, this study supports the safety of non-operative ELS management.

Endovascular treatment of pulmonary sequestration with thoracic endograft: Two case reports.

INTRODUCTION: Pulmonary sequestration (PS) is a rare congenital lung malformation. The classical treatment of the disease has been conventional surgery with resection of abnormal parenchyma. Recently, the endovascular embolization has been proposed for the treatment of this disease. Here we present 2 cases of PS successfully treated with thoracic endograft. PATIENT CONCERNS: Two patients with abnormal consolidation in the left lower lobe were admitted in our hospital. DIAGNOSIS: Chest computed tomography angiography (CTA) showed abnormal consolidation in the left lower lobe, which received systemic blood supply from the descending aorta in both patients. So the diagnosis of PS was confirmed. INTERVENTIONS: Endovascular treatment with thoracic endograft was successfully performed. OUTCOMES: The patients recovered well and were completely free of symptoms. And the CTA follow-up showed the abnormal pulmonary parenchyma shrunk significantly. CONCLUSIONS: Endovascular treatment with thoracic endograft is a promising treatment option for PS.

Congenital lung lesions.

Diagnosis and management of congenital lung malformations has evolved dramatically over the past several decades. Advancement in imaging technology has enabled earlier, more definitive diagnoses and, consequently, more timely intervention in utero or after birth, when indicated. These advancements have increased overall survival rates to around 95% from historical rates of 60%. However, further refinement of diagnostic technique and standardization of treatment is needed, particularly as the increased sensitivity of diagnostic imaging results in more frequent diagnoses. In this article, we provide an updated review of the diagnostic strategies, management, and prognosis of congenital lung malformations.

Coil embolization of intralobar pulmonary sequestration - an alternative to surgery: a case report.

BACKGROUND: Pulmonary sequestration is a congenital lung disease characterized by nonfunctioning pulmonary tissue that lacks normal communication with the bronchial tree and is supplied by a nonpulmonary systemic artery. Symptomatic bronchopulmonary sequestration is uncommon, seen more frequently in the pediatric population than in adults. It has traditionally been treated with surgical resection; however, a limited but growing number of cases have been treated with angiographic embolization. Given the inherent risks of cardiothoracic surgery, embolization of the anomalous vessel is an enticing alternative treatment. We present a case of a 56-year-old woman with known, symptomatic, intralobar pulmonary sequestration that was successfully treated with coil embolization. CASE PRESENTATION: A 56-year-old Pacific Islander woman with a history of chronic myeloid leukemia was admitted to the hospital with an episode of hemoptysis. Computed tomography of the chest demonstrated left lower lobe intralobar pulmonary sequestration fed by a large tortuous vessel branching off of the descending thoracic aorta. Surgical resection of the sequestration is the current standard treatment strategy of symptomatic intralobar pulmonary sequestration. The cardiothoracic surgeon noted that given the size and location of arterial blood supply, intervention would involve thoracotomy and lobectomy. The interventional radiologist offered embolization of the lesion as an alternative to surgery. Multiple coils, 6-13 mm in size, were used to embolize the sequestration. No considerable flow distal to the coils was noted postembolization. CONCLUSIONS: Intralobar pulmonary sequestration is a rare condition that typically requires surgical management. This case demonstrates the efficacy of coil embolization as an alternative management strategy. To date, limited case reports of adults treated with endovascular embolization exist. Treatment of symptomatic pulmonary sequestration with embolization can be considered as an alternative to surgical resection.

[Cardiovascular, broncho-parenchimal and neoplastic anomalies related to pulmonary sequestration: a critical review.] / Anomalie cardiovascolari, bronco-parenchimali e neoplastiche connesse con la sequestrazione polmonare: rassegna critica.

The main aim of this critical review on the pulmonary sequestration is to focus the attention of pneumologists, thoracic surgeons and oncologists on some features concerning the heart, vessels, lungs, pleura, diaphragm as well as unusual infections and neoplasms, closely related to such rare disease. The Author emphasizes the importance of these aforementioned peculiarities with reference to the diagnosis timelines as well as to treatment appropriateness. He believes that the new advanced tools to day available makes to achieve this target: it is a must to take in more greater account the rare and insidious pulmonary sequestration at least in the context of post-graduate specialty courses.

Hybrid surgery in treatment of pulmonary sequestration with abdominal aorta feeding vessel: a case report.

BACKGROUND: Pulmonary sequestration is a rare congenital pulmonary dysplasia, which requires surgical resection (either via open thoracotomy or video-assisted thoracoscopic surgery [VATS] or via endoluminal occlusion of the abnormal feeding vessel). CASE PRESENTATION: We described a 51-year-old female patient with a history of recurrent cough and repeated pneumonia. She was referred to our hospital for further work-up of pulmonary sequestration. We performed a hybrid surgery (i.e., embolization of the aberrant feeding vessel of the sequestration combined with wedge resection of the left lower lobe lesion through VATS). The patient was discharged on the sixth postoperative day in good condition and without complications. CONCLUSIONS: We believe that a hybrid operation is safer, more feasible, and more comprehensive than other treatments.

Prenatal growth characteristics and pre/postnatal management of bronchopulmonary sequestrations.

PURPOSE: The prenatal natural history of intralobar and extralobar bronchopulmonary sequestrations (BPSs), including lesion growth patterns and need for prenatal intervention, have not been fully characterized. We review our series of BPSs to determine their natural history and outcomes in the context of the need for prenatal intervention. METHODS: A retrospective review of the pre/postnatal course of 103 fetuses with an intralobar (n=44) or extralobar BPS (n=59) managed at a single institution between 2008 and 2015 was performed. Outcomes included prenatal lesion growth trajectory, presence of hydrops, need for prenatal intervention, survival, and postnatal surgical management. RESULTS: Most extralobar (71%) and intralobar BPSs (94%) decreased in size or became isoechoic from initial to final evaluation. Peak lesion size occurred at 26-28weeks gestation. Eight fetuses developed hydrothorax, four of which (all extralobar BPSs) also developed hydrops. All four hydropic fetuses received maternal betamethasone, and three hydropic fetuses underwent thoracentesis and/or thoracoamniotic shunt placement with subsequent hydrops resolution. All fetuses survived. Forty-one intralobar (93%) and 35 extralobar BPSs (59%) were resected after birth. CONCLUSIONS: BPSs tend to decrease in size after 26-28weeks gestation and rarely require fetal intervention. Lesions resulting in hydrothorax ± hydrops can be effectively managed with maternal steroids and/or drainage of the hydrothorax. LEVEL OF EVIDENCE: IV.

Outcome of Bronchopulmonary Sequestration with Massive Pleural Effusion after Intrafetal Vascular Laser Ablation.

OBJECTIVE: To assess the outcome of 12 fetuses with bronchopulmonary sequestration (BPS) and massive pleural effusion after intrafetal vascular laser ablation (VLA). METHODS: All fetuses with BPS and massive pleural effusion that were treated with intrafetal VLA during a 5-year period (2012-2016) were reviewed for safety, intrauterine course, and postnatal outcome. RESULTS: In the study period, 12 fetuses with BPS were treated with VLA. In 7 (58.3%) fetuses, complete cessation of blood flow was achieved after the first VLA, while in 5 (41.7%) fetuses, residual perfusion of the feeding vessel was demonstrated at follow-up. A second intervention was successful in 4 of 5 (80%) fetuses. Overall, in 11 of 12 (91.7%) fetuses, complete coagulation of the feeding vessel could be achieved, followed by a reduction in size or complete resolution of the BPS. All 11 fetuses with successful prenatal intervention were live-born at a median gestational age of 39+1 (range, 37+5-41+2) weeks. Postnatally, 2 (18.2%) of the 11 newborns underwent sequestrectomy, as well as the preterm newborn on which a second fetal intervention was not feasible. CONCLUSION: VLA is an effective and safe treatment of BPS that appears to be of benefit in improving prognosis and decreasing the need for postnatal sequestrectomy.

Long-term outcomes of congenital lung malformations.

Congenital lung malformations comprise a group of anatomical abnormalities of the respiratory tree including congenital cystic malformations, bronchopulmonary sequestrations, bronchogenic cyst, bronchial atresia, and congenital lobar emphysema. These anomalies are detected with increasing frequency by pre-natal sonography, but may also present for the first time with symptoms in childhood or later life. When symptomatic, there is little controversy that resection is indicated, which is usually curative. When a lesion is asymptomatic there is greater debate regarding the benefit of resection versus continued observation. This article provides an overview of the spectrum of disorders, the management options available and the long-term outcomes associated with each treatment option.

[Endovascular embolization in the treatment of the pulmonary intralobar sequestration]. / Embolización endovascular en el tratamiento del secuestro pulmonar intralobar.

INTRODUCTION: Up to date the classical therapeutic approach for intralobar sequestrations (IS) has been surgical excision. However, systemic arteries embolization is presented as an alternative, and even constitutes the first line of treatment in some centers. We summarize our experience in selected cases with the aim of preserving the maximum lung parenchyma. MATERIAL AND METHODS: Retrospective study of IS who underwent endovascular embolization at our institution between 2013 and 2014. RESULTS: Three patients of 12, 14 and 21 months old were treated. Two patients had unilateral IS, one in the left lower lobe (LLL) and the other in the right lower lobe (RLL); the third one had bilateral lesions (a CAM-S complex in the RLL and a IS in the LII). The embolization of the four lesions was performed via right femoral artery. The case with bilateral lesions underwent thoracoscopic lobectomy of the CAM-S 7 months after embolization, finding adherences to the diaphragm. None of the patients had immediate complications and were discharged within 48 hours after embolization. In successive ultrasound and plain radiographs controls, with a follow-up of 6, 18 and 30 months, no complications were found. CONCLUSIONS: Systemic vessels embolization is a treatment option for the treatment of IS that avoids surgery, preserves lung parenchyma and does not preclude surgical resection in case of treatment failure or presence of parenchymal lesions. Longer follow-up is needed to determine long-term effectiveness.

INTRODUCCION: El tratamiento de elección de los secuestros intralobares (SI) es la lobectomía pulmonar. La mayoría de complicaciones se asocian a la vascularización sistémica pulmonar anómala. La embolización de las arterias sistémicas se presenta como una alternativa terapéutica, que incluso constituye la primera línea de tratamiento en algunos centros. Resumimos nuestra experiencia al aplicar esta técnica en casos seleccionados. MATERIAL Y METODOS: Estudio retrospectivo de los casos con SI tratados en nuestro centro mediante embolización entre 2013 y 2014, centrado en las indicaciones, resultados, complicaciones y seguimiento. Siendo la lobectomía la primera línea de tratamiento la embolización se reservó para casos seleccionados: bilaterales, con el objetivo de preservar parénquima pulmonar o ante el rechazo familiar a la intervención quirúrgica. RESULTADOS: Se trataron tres pacientes de 12, 14 y 21 meses. Dos presentaban un SI unilateral, uno en lóbulo inferior izquierdo (LII) y otro en el lóbulo inferior derecho (LID); el tercero lesiones bilaterales (un SI en LID y un complejo MAQ-secuestro en LII). Se embolizaron las cuatro lesiones por punción de arteria femoral derecha. El caso con lesiones bilaterales fue intervenido a los 7 meses tras la embolización, realizándose una lobectomía toracoscópica del complejo MAQ-secuestro, hallando adherencias al diafragma. Ninguno presentó complicaciones inmediatas y fueron dados de alta a las 48 horas. En los controles realizados con ecografía y radiografías simples, con seguimientos de 31, 47 y 56 meses, no se han observado complicaciones clínicas ni radiológicas. CONCLUSIONES: La embolización de los vasos sistémicos es una opción en el tratamiento del SI que permite evitar una cirugía, conservar parénquima pulmonar y no impide la exéresis quirúrgica en caso de fallo del tratamiento o lesión parenquimatosa. Es preciso un mayor seguimiento para determinar la efectividad a largo plazo.

Antenatally Detected Congenital Pulmonary Airway Malformations: The Oxford Experience.

Congenital airway pulmonary malformations are increasingly being diagnosed, but their management continues to remain controversial. Our approach has been to offer surgery to mitigate the risk of infection and possible malignancy. All patients routinely undergo a CT scan of the chest postnatally and once the diagnosis is confirmed, minimal access surgery is offered. Our anesthetists provide single-lung ventilation to enhance the operative view. We conducted a retrospective review over a 10-year period, during which 91 patients were prenatally suspected to have a cystic lung lesion. There were 88 live births of which 29 (33%) cases were initially managed conservatively based on CT findings. Five of these patients, however, became symptomatic needing surgery. A total of 64 (73%) patients underwent surgery with the most common lesions being congenital pulmonary airway malformations (CPAMs) (24), hybrid lesions (19), and pulmonary sequestrations (12). The median age at surgery was 5 months (1 day to 17 months). Using a minimal access approach, 41 (64%) cases were completed with 17 performed open from the onset. Open surgery was indicated in neonates who became symptomatic within the first few weeks of life as well as patients in respiratory distress that would not tolerate either single-lung ventilation or gas insufflation. There were six further conversions to open from minimal access surgery due to poor visualization or technical difficulties. One patient needed a perioperative blood transfusion and one patient had a more prolonged stay due to persistent air leak managed conservatively. Among asymptomatic patients, evidence of microscopic disease was seen, which included infection as well as two cases of tumors, one pleuropulmonary blastoma seen as part of a CPAM, and one rhabdomyomatous dysplasia seen in the CPAM component of a hybrid lesion. In our experience, excising asymptomatic lesions is safe with minimal complications. Single-lung ventilation in combination with thoracoscopy provides excellent vision. There is a risk of infection and a definite, albeit low, risk of malignancy, which may outweigh the benefits of conservative management.