Impact of the bronchopulmonary sequestration on endobronchial tuberculosis: the case report and the review of literature

Introduction: We describe the rare case of endobronchial tuberculosis (EBTB) and chronic pulmonary atelectasis with mediastinal distortion. Finding of the concomitant venous anomaly of inferior vena cava revealed the diagnosis of bronchopulmonary sequestration. Case Report: A 22-year-old Caucasian woman presented with a history of chronic cough, initially treated as bronchial asthma for a year. Chest X-ray showed fibrocaseous cavernous tuberculosis on the right lung. Acid Fast Bacilli (AFB) were found in sputum samples. Patient was treated for 6 months with usual antituberculous regiment. Control chest X-ray showed subatelectasis of the upper right lobe. Six months later the first thorax computed tomography (CT) showed complete atelectasis of the right lung. Patient was admitted to the hospital again after 6 years due to the persistent fever and cough. Endoscopic finding and histopathological analysis confirmed EBTB. Thoracic CT scan revealed duplication of inferior vena cava which led to profound vascular analysis and aberrant arterial vascularization of aortic origin that contributed to the diagnosis of bronchopulmonary sequestrations. Antituberculous treatment was initiated (streptomycin, isoniazid, rifampicin, ethambutol and pyrazinamide) and lasted for 8 months. After 8 months a follow-up fiberoptic bronchoscopy showed the progression of endoscopic finding with 60-70% tracheal stenosis. Histopathological finding of the mid-trachea showed non-specific granulations. During 7 years of follow-up repeated bronchoscopy and thoracic CT scans were unchanged and patient was well-shaped. Conclusions: The clinician should consider bronchopulmonary sequestration in the cases of recurrent EBTB.

ENDOSURGICAL TECHNOLOGIES FOR TREATMENT OF BRONCHOPULMONARY SEQUESTRATION IN CHILDREN

The study systematized the long-term experience of thoracoscopic and laparoscopic treatment of intra- and extrapleural forms of bronchopulmonary sequestration (BPS). Materials and methods of research: a retrospective study analysed clinical data of 20 patients with BPS who have underwent thoracoscopic and laparoscopic surgeries for 15 years, starting from 2005. The main surgical methods included thoracoscopic lobectomy, thoracoscopic sequestrectomy, laparoscopic sequestrectomy and combined thoracoscopic transdiaphragmatic access in a patient with multiple forms of BPS. Results: a total of 20 patients with BPS had an intralobar form, 5 had an extralobar form, 4 had an extrapleural type, and 1 had a combined (extralobar + extrapleural) form. Intralobar form of BPS was found in the left lower lobe in 6 patients, in the right lower lobe – in 4. Extralobar BPS was found near the left lower lobe in 3 patients, near the lower right lobe – in 2. Extrapleural BPS was observed in the abdominal cavity under the diaphragm in 3 patients, in one patient – in the thickness of the diaphragmatic muscle. One patient had a combined form of BPS in the form of an association of extralobaric and intra-abdominal types. Thoracoscopy was performed in 16 patients, laparoscopy in 4 patients. The mean operative time was 60 min (range: 40–100 min). Intraoperative blood loss was not observed. A drainage tube was installed in 10 patients with intralobaric BPS. In all other cases, the pleural and abdominal cavity was not drained. A histological examination of all resected pulmonary sequesters showed that in half of the cases (10/20) the anomaly was associated with other malformations of the lungs (cystoadenomatous malformation of the lungs, bronchogenic cyst or intestinal duplication). During the 5 year average observation period after surgery (range: 1–15 years), musculoskeletal deformities of the chest and adhesive intestinal obstruction were not recorded. Conclusion: the series of minimally invasive treatment of patients with BPS presented in this study demonstrates the effectiveness and safety of this approach, especially for the surgical correction of those forms of pulmonary sequestration when they are located in hard-to-reach anatomical spaces of the body.

Rare cause of haemoptysis: bronchopulmonary sequestration

Bronchopulmonary sequestration is a rare congenital pulmonary abnormality of the lower airways, which includes an abnormal and non-functioning lung tissue not communicating with the tracheobronchial tree and having aberrant blood supply from systemic circulation with variable venous drainage. The incidence of sequestration is around 0.15%–6.4% of all congenital lung malformations.Common presenting features are cough and expectoration. Misdiagnosed cases may present with recurrent infections and haemoptysis. CT of the chest with contrast is the imaging modality of choice.This is a case report of a 32-year-old woman who presented with cough and haemoptysis. CT of the chest showed a multiloculated mass-like lesion in the left lower lobe with a feeding artery from coeliac plexus and venous drainage via the normal left pulmonary vein.Based on CT chest findings, diagnosis of intralobar pulmonary sequestration was made. The patient was reviewed by cardiothoracic surgeons and underwent surgical resection of the sequestrated lung.Common presenting features are cough and expectoration. Misdiagnosed cases may present with recurrent infections and haemoptysis. CT of the chest with contrast is the imaging modality of choice.

Non-immune hydrops fetalis complicating bronchopulmonary sequestration: A case report

Background: Non-immune hydrops (NIH) associated with bronchopulmonary sequestration (BPS) is quite rare with a 95% risk of intrauterine fetal death, without a fetal intervention. Case Presentation: We describe a case of an antenatally diagnosed extralobar BPS with severe NIH, who underwent fetal thoracentesis, but had worsening of NIH requiring an emergency cesarean section. Postnatally, the baby required skillful intensive care management and timely surgical management in the form of a sequestrectomy. Conclusion: We report the smallest neonate with antenatally diagnosed NIH complicating BPS treated successfully by early neonatal surgery.

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

<b><i>Objective:</i></b> 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. <b><i>Methods:</i></b> 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. <b><i>Results:</i></b> 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%. <b><i>Conclusion:</i></b> 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.

Pulmonary Sequestration Associated with Actinomycosis: A Case Report

Background: Bronchopulmonary sequestration is a rare congenital malformation of the lower respiratory tract; it consists of a nonfunctioning mass of lung tissue that is irrigated by an anomalous systemic artery. The association with Actinomyces superinfection has not been well established. Methods: We present the case of a 35-year-old woman with a history of recurrent episodes of pneumonia. Based on radiological and histopathological examination, she was diagnosed with intralobar bronchopulmonary sequestration associated with Actinomyces infection. Promoting clinical suspicion is essential to diagnose pulmonary actinomycosis in patients with recurrent pneumonia, to improve early recognition and timely management.