Intralobar sequestration of lung, associated with foregut diverticulum (qesophagobronchial fistula) and an aberrant artery

AbstractWe describe an infant of 8 months who presented with left ventricular dilation due to an extensive intralobar sequestration of the right lung. The pulmonary sequestration was associated with a patent arterial duct and a right aortic arch. Percutaneous closure of the anomalous aberrant artery feeding the sequestrated lung resulted in prompt regression of the left ventricular enlargement.

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Successful thoracoscopic treatment for tracheoesophageal fistula and esophageal atresia of communicating bronchopulmonary foregut malformation group IB with dextrocardia: a case report of VACTERL association

Surgical Case Reports ◽

10.1186/s40792-020-01099-y ◽

2021 ◽

Vol 7 (1) ◽

Author(s):

Toshio Harumatsu ◽

Tatsuru Kaji ◽

Ayaka Nagano ◽

Mayu Matsui ◽

Masakazu Murakami ◽

...

Keyword(s):

Tracheoesophageal Fistula ◽

Thoracoscopic Surgery ◽

Anastomotic Site ◽

Vacterl Association ◽

Lower Esophagus ◽

Intralobar Sequestration ◽

Working Space ◽

Esophageal Anastomosis ◽

Cardiac Condition ◽

Foregut Malformation

Abstract Background A communicating bronchopulmonary foregut malformation (CBPFM) group IB is very rare congenital malformation. Group IB is associated with tracheoesophageal fistula and esophageal atresia (TEF-EA) and a portion of one lung arisen from the esophagus (Gerle et al. in N Engl J Med. 278:1413–1419, 1968). The coexistence of TEF-EA and dextrocardia is also a rare and challenging setting for repair of TEF-EA. Therefore, the thoracoscopic surgery for TEF-EA require the technical devise because of the small operative space. We herein report a rare case of CBPFM group IB with intralobar sequestration of lung and a successful performing of thoracoscopic surgery for EA with dextrocardia in VACTERL association. Case presentation A 2.2-kg term male neonate was born with an anal atresia, coarctation of the aorta, TEF-EA, renal anomalies, radial hemimelia, limb abnormalities (VACTERL association) and hypoplasia of the right lung with dextrocardia. The patient developed respiratory distress after admission. A two-stage operation for the TEF-EA was planned because of multiple anomalies and cardiac condition. In the neonatal period, esophageal banding at the gastroesophageal junction and gastrostomy were performed to establish enteral nutrition. After gaining body weight and achieving a stable cardiac condition, thoracoscopic surgery for TEF-EA was performed. The thoracoscopic findings revealed a small working space due to dextrocardia. To obtain a sufficient working space and to perform secure esophageal anastomosis, an additional 3-mm assistant port was inserted. To close the upper and lower esophagus, anchoring sutures of the esophagus were placed and were pulled to suspend the anastomotic site. Esophageal anastomosis was successfully performed. An esophagogram after TEF-EA surgery showed the connection between the lower esophagus and right lower lung. The definitive diagnosis was CBPFM group IB with intralobar sequestration. The thoracoscopic surgery was performed again for establishing oral intake. After transection of the bronchoesophageal fistula, the patient could perform oral feeding without pneumonia or respiratory distress. Conclusions CBPFM type IB with intralobar sequestration is a rare condition. CBPFM type IB should be considered for a patients with respiratory symptom after radical operation for TEF-EA. In the present case, suspending the anastomotic site was effective and useful in thoracoscopic surgery for a TEF-EA patient with dextrocardia.

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Cystic Adenomatoid Change Is Common to Various Forms of Cystic Lung Diseases of Children

Archives of Pathology & Laboratory Medicine ◽

10.5858/2002-126-0934-cacict ◽

2002 ◽

Vol 126 (8) ◽

pp. 934-940

Author(s):

Yukihiro Imai ◽

Eugene J. Mark

Keyword(s):

Lung Disease ◽

Blood Vessels ◽

Bronchial Tree ◽

Congenital Cystic Adenomatoid Malformation ◽

Cystic Lesions ◽

Cystic Lung ◽

Intralobar Sequestration ◽

Cystic Adenomatoid Malformation ◽

Lobar Emphysema ◽

Bronchial Atresia

Abstract Context.—Congenital cystic adenomatoid malformation, intralobar sequestration, extralobar sequestration, bronchopulmonary foregut malformation, bronchial atresia, and lobar emphysema generally are treated as separate entities, but there are commonalities among them. Objective.—To clarify the identity of congenital cystic adenomatoid malformation in relation to other cystic diseases. Design.—We studied 10 consecutive cases of surgically resected cystic lung disease in children. We studied the bronchial and vascular trees by mapping bronchi, pulmonary blood vessels, systemic blood vessels, the size and configuration of cysts, and associated scarring. Results.—The cystic lesions proved to be the following: 1 case of congenital cystic adenomatoid malformation, 2 cases of bronchial atresia with congenital cystic adenomatoid malformation, 1 hybrid case of congenital cystic adenomatoid malformation and intralobar sequestration, 1 case of intralobar sequestration with an aberrant hilum, 3 cases of intralobar sequestration with bronchial obliteration, 1 case of lung abscess, and 1 case of lobar emphysema. Congenital cystic adenomatoid malformation was the sole pathologic diagnosis in 1 case, but 7 other specimens had cystic adenomatoid change to various degrees. There was no bronchial connection between the cystic lesions and the patient's airway in 8 cases. The bronchial tree was absent in the cystic lung in 2 cases. Bronchus tapered into scar near the cystic lesion in 4 cases. Only 1 case had no significant bronchial abnormality. Conclusion.—The high incidence of cystic adenomatoid change in cystic lung disease associated with an abnormality of the bronchial tree suggests that cystic adenomatoid change may develop together with and be related to other congenital or acquired conditions in the lung.

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Recurrence of right lower lobe pneumonia 3 years after the first episode in an otherwise healthy 13-year-old girl

Monaldi Archives for Chest Disease ◽

10.4081/monaldi.2017.802 ◽

2017 ◽

Vol 87 (1) ◽

Author(s):

Antonino Capizzi ◽

Oliviero Sacco ◽

Giovanni A. Rossi ◽

Michela Silvestri

Keyword(s):

Lower Lobe ◽

Histological Evaluation ◽

First Episode ◽

Recurrent Pneumonia ◽

Surgical Specimen ◽

Lung Lobe ◽

Intralobar Sequestration ◽

Systemic Artery ◽

Contrast Enhanced Ct ◽

Diagnostic Work

<p>Recurrent pneumonia is one of the most frequent reasons for referral to paediatric chest physicians. The diagnostic work-up is dependent on whether infection repeatedly occurs in the same lung lobe, or affects multiple lobes and/or different areas in different episodes. A 13-year-old girl was admitted with a second episode of right lower lobe pneumonia. The chest x-ray demonstrated an inhomogeneous opacity, without a clearly recognizable segmental distribution. A contrast-enhanced CT scan, was therefore performed that showed a polycyclic consolidation with blood supply from a systemic artery, originated from the thoracic aorta. A diagnosis of superinfection of an intralobar sequestration was made. The patient was treated with systemic antibiotics and, four weeks later, a segmental resection of the lesion was performed. The histological evaluation of the surgical specimen confirmed the diagnosis. </p>

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