Two cases of prenatally diagnosed congenital lobar emphysema caused by lobar bronchial atresia

Seo, Takahiko; Ando, Hisami; Kaneko, Kenitiro; Ono, Yoshio; Tainaka, Takahisa; Sumida, Wataru; Hayakawa, Masahiro; Itakura, Atsuo

doi:10.1016/j.jpedsurg.2006.08.037

Cited by 34 publications

(29 citation statements)

References 13 publications

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“…Airway obstruction results in a dysplastic response of the bronchial tree distal to the obstruction, which becomes filled with mucus and forms a bronchocele [10,11,12,13]. Clinical relevance depends on the amount of lung parenchyma involved.…”

Section: Discussionmentioning

confidence: 99%

Decompression through Tracheobronchial Endoscopy of Bronchial Atresia Presenting as Massive Pulmonary Tumor: A New Indication for Fetoscopic Surgery

et al. 2012

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This is the first report of fetoscopy to successfully treat a case of bilobar bronchial atresia presenting in the form of a massive right lung tumor causing severe thoracic compression with mediastinal shift, pulmonary hypoplasia and eventually fetal hydrops. The presence of an echolucent tubular structure in the pulmonary hilum was identified as the intrapulmonary continuation of the interrupted main bronchus, and led to the diagnosis of main bronchial atresia. After extensive discussion and counseling, a fetoscopic operation was designed and indicated. The procedure consisted of a fetal tracheoscopy and access to the right main bronchus, which was perforated with a diode laser until a communication with the dead end of the intrapulmonary main bronchus was achieved. After the operation a significant reduction in size was observed, hydrops and mediastinal shift disappeared and O/E LHR increased to 85%. The fetus was born at term and lobectomy of two dysplastic pulmonary lobes was performed. The infant is developing well at 14 months of age. This report demonstrates that fetoscopic airway decompression can achieve fetal survival in the rare event of main bronchus atresia.

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Section: Discussionmentioning

confidence: 99%

Decompression through Tracheobronchial Endoscopy of Bronchial Atresia Presenting as Massive Pulmonary Tumor: A New Indication for Fetoscopic Surgery

et al. 2012

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“…Seo et al [12] outlines two cases of congenital lobar emphysema that have been caused by lobar bronchial atresia. In cases in which CLE is directly linked to bronchial atresia, the pulmonary parenchyma distal to the lesion may be aerated by collateral ventilation [12]. This is not as effective in expiration as it is in inspiration, resulting in a significant amount of air trapping.…”

Section: Summary and Discussionmentioning

confidence: 99%

“…The pathogenesis has been described as "localized interruption of the blood supply to an already formed bronchus, resulting in bronchial infarction and luminal obstruction, or developmental abnormalities of bronchial cartilage after completion of bronchial branching [13]. In patients in whom congenital lobar emphysema is caused by bronchial atresia, the affected emphysematous lobe is extremely large, with concomitant compression of the normal lung tissue, displacement of the mediastinum, and herniation of the emphysematous lobe across the antrerior mediastinum into the opposite side of the chest [12]. These findings are consistent with the most typical presentations of CLE.…”

Section: Summary and Discussionmentioning

confidence: 99%

Congenital Lobar Emphysema in Adult Treated With Video Assisted Thoracoscopic Lobectomy

Perry¹,

Espiritu²,

Schroeder³

2013

J Pulm Respir Med

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Congenital Lobar Emphysema (CLE) is an uncommon pulmonary disorder in which one or more pulmonary lobes is over-inflated and distended. Fewer than 5% of cases are diagnosed beyond the first 6 months of life. We describe CLE that presented in a 25 yr old female who was successfully treated with minimally invasive video assisted thoracoscopic surgery (VATS). Case ReportA 25-year old Puerto Rican female presented with increased shortness of breath, cough and chest pain. Her past medical history included diagnosis of asthma In Puerto-Rico at the age of five, treated with advair. During her third pregnancy, at the age of 23, the patient experienced shortness of breath and reported "fainting". A chest CT at that time diagnosed her with lobar emphysema.In the United States, she presented to an outside hospital with exacerbation of shortness of breath, where she was thought to have a pneumothorax, and a right chest tube was placed. She was then transferred to the Medical Center ICU. At presentation, She was afebrile, exhibiting tachycardia (117 bpm), with 28 respirations per minute, oxygen saturation of 91%, and decreased breath sounds on the right chest field.Pulmonary function test suggested of Restrictive Lung Disease (RLD) ( Table 1). The patient's initial X-rays, as seen in Figure 1, indicate hyperlucency on the right side, flattening of the right diaphragm, and a slight shift of mediastinum to the left. The right lung is collapsed inferiorly. There are a severe number of bullae within the right hemithorax, with some consolidation or collapse in the region of the right perihilar region.CT taken before surgery documented large bullae, mediastinal shift, and compressed right lung. (Figure 2 and 3). Treatment and Surgical FindingsSince the patient's shortness of breath was not successfully managed with Oxygen support and chest tube drainage, she was scheduled for a bronchoscopy and a Video Assisted Thoracoscopic Surgery (VATS) -right upper lobectomy. On BronchoscopyThe Right Upper Lobe (RUL) takeoff was a slit-like lumen, slightly distorted -confirms the diagnosis of CLE.VATS right upper lobectomy was performed with general endotracheal anesthesia with a double lumen tube and one lung ventilation. We used 3 ports; 10mm port in the seventh intercostal space in anterior axillary line, 10mm trocar in posterior axillary Using a 10 mm 30 degree Olympus scopeThe right upper lobe composed entirely of large bullae. The bullae did not deflate and were densely adherent to the chest wall. There were also aberrant blood vessels from the chest wall to the bullae, with no apparent superior pulmonary vein, pulmonary artery, or bronchi going to the bullous portion. However, the segmental bronchi and major pulmonary vessels were found attached to the area of the hilum of the atritic lobe. An endoscopic EndoGIA (Covidian) used to divide the vessels and the rudimentary bronchus. The lobe removed with an endobag. A 28 french chest tube was left in the chest. PathologyMicroscopic examination of the resected lobe showed dense fi...

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“…Extrinsic causes include compression of bronchus from outside by vascular rings or lymph nodes. (2), (3) Bronchial atresia and defective bronchial cartilage are common causes. (3),(4), (5) Exact cause of CLE is unknown in 50% of the cases.…”

Section: Introductionmentioning

confidence: 99%

“…(2), (3) Bronchial atresia and defective bronchial cartilage are common causes. (3),(4), (5) Exact cause of CLE is unknown in 50% of the cases. (6) We report our experience in 19 cases of CLE discussing their clinical features along with histology.…”

Section: Introductionmentioning

confidence: 99%

Congenital Lobar Emphysema, a Histopathological Study

O.H¹,

Geetha²,

Mandakini³

et al. 2015

jemds

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Congenital lobar emphysema is a very rare congenital cystic malformation of the lung that can cause acute respiratory distress early in life. This study reviews 19 cases of CLE over a period of 5 years at a Tertiary Pediatric Referral Center. The cases were studied with special emphasis on Histopathological findings of the lobectomy specimens, particularly bronchial cartilage abnormalties, radial alveolar count and their predictive value in pathogenesis of CLE. This condition can mimick other causes of respiratory distress in the neonatal period. Early diagnosis and effective surgical management is curatve.

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Two cases of prenatally diagnosed congenital lobar emphysema caused by lobar bronchial atresia

Cited by 34 publications

References 13 publications

Decompression through Tracheobronchial Endoscopy of Bronchial Atresia Presenting as Massive Pulmonary Tumor: A New Indication for Fetoscopic Surgery

Decompression through Tracheobronchial Endoscopy of Bronchial Atresia Presenting as Massive Pulmonary Tumor: A New Indication for Fetoscopic Surgery

Congenital Lobar Emphysema in Adult Treated With Video Assisted Thoracoscopic Lobectomy

Congenital Lobar Emphysema, a Histopathological Study

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