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11/19. Bronchopulmonary foregut malformations. A unifying etiological concept.

    Two cases of congenital bronchopulmonary foregut malformation are reported and another 27 cases reviewed and the findings analyzed. The left lower lobe and the entire right lung are the most common locations of pulmonary involvement. The distal portion of the esophagus or cardioesophageal junction was the most common site of communication (83%). The majority of the patients (60%) presented in the first eight months of life, and the incidence in females was nearly twice that in males. Chronic cough, recurrent pneumonias and respiratory distress were the most common clinical findings, whereas two patients were totally asymptomatic. The esophagogram was the single most useful diagnostic procedure (82%). The microscopic structure of the congenital fistula resembled esophagus, bronchus or both. Surgical treatment was curative in most instances. Sime deaths occurred prior to corrective surgery, whereas the postoperative deaths in most instances were related to severe associated congenital anomalies. We believe a common embryologic pathogenesis leads to the formation of a variety of bronchopulmonary foregut malformations. These bronchopulmonary foregut malformations include intralobar and extralobar sequestrations, pulmonary sequestration with patent, or involuted--partial or complete--gastroesophageal communication, esophageal or gastric diverticula, and esophageal or bronchogenic duplication cysts.
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12/19. Congenital bronchopulmonary foregut malformations. Intralobar and extralobar pulmonary sequestrations communicating with the foregut.

    Two unusual variants of bronchopulmonary foregut malformations are presented. The first case was that of a 12-year-old male with a history of pectus excavatum in whom severe lobar emphysema developed secondary to an intralobar pulmonary sequestration that communicated with the esophagus. This case was unusual in that foregut communications and associated congenital anomalies are generally believed to be restricted to extralobar pulmonary sequestrations. The second case was that of a 27-year-old woman with an extralobar pulmonary sequestration that communicated with the esophagus. The sequestration was unusual in that it arose in the anterior mediastinum and received the bulk of its blood supply from the pulmonary artery. The occurrence of mixed forms of pulmonary sequestrations supports the hypothesis that extralobar and intralobar sequestrations and sequestrations with foregut communication are related thoracic disorders that are best considered bronchopulmonary foregut malformations.
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13/19. A case of intralobar pulmonary sequestration with high serum CA19-9 levels.

    A 38-year-old female presented with cough and fever. A chest X-ray examination revealed an abnormal shadow in the posteroinferior portion of the left hemithorax, and a laboratory examination showed that the serum carbohydrate antigen 19-9 (CA19-9) level was markedly high (1000 U/ml). A left thoracotomy showed an intralobar pulmonary sequestration of the left lower lobe, and after a left lower lobe lobectomy, the serum level of CA19-9 decreased to normal. Increased CA19-9 activity was detected by immunohistochemistry in the epithelia of bronchioles in the pulmonary sequestration. This communication is the first to report a case of increased activity of CA19-9 in pulmonary sequestration.
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14/19. Pulmonary sequestration with congenital gastroesophageal communication. Report of two cases.

    Two children with pulmonary sequestration and gastroesophageal connections are described. barium studies demonstrated the site of the connection and angiography showed the vascular connections enabling successful surgery to the performed.
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15/19. Malformation of bronchopulmonary foregut with systemic and pulmonary arterial blood supply.

    A case of congenital malformation of the bronchopulmonary foregut with communication between the esophagus and sequestered lobe is reported in a six-month-old boy. Only 29 similar cases have been reported previously, and this case was especially unusual in that the communication was from the middle portion of the esophagus to a right apical sequestration. Another unusual feature was that the sequestered segment was supplied by four systemic arteries from the thoracic aorta, as well as by branches from the right pulmonary artery. Within the sequestration, there was shunting of blood from the systemic to pulmonary arterties with reversal of flow in the pulmonary arterial branches.
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16/19. bronchopulmonary sequestration as a rare cause of acute, massive intraesophageal bleeding.

    We have described a 69-year-old woman with sequestered segment of lung located in the right upper lobe, with open communication with the esophagus. Investigation of the acute anemia produced by acute blood loss into the gastrointestinal tract led to the diagnosis and treatment of this rare condition.
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17/19. Spectrum of pulmonary sequestration: association with anomalous pulmonary venous drainage in infants.

    Pulmonary sequestration is a spectrum of related lesions, each of which may be absent or present: (1) bronchial sequestration of pulmonary parenchyma; (2) arterial supply from systemic circulation; (3) anomalous pulmonary venous drainage to the right atrium; (4) communications between bronchus and esophagus; (5) defects of diaphragm; (6) gross lung anomalies, such as horseshoe lungs or hypoplasia. Any combination of these primary lesions can occur in an individual patient. diagnosis should be directed towards each component of the spectrum. Of special importance is the venous connection, as anomalous pulmonary venous drainage can involve not only the sequestered segment but the entire ipsilateral lung, making surgical therapy far more complex. Treatment of choice is surgical resection, associated, if needed, with rerouting of the pulmonary venous return. classification of sequestration of the lung as intra- and extralobar is of secondary importance: these 2 groups do not represent lesions of different embryological significance.
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18/19. mycetoma within an intralobar sequestration. Evidence supporting acquired origin for this pulmonary anomaly.

    A rare example of an intralobar sequestration (ILS) containing a fungal mycetoma is reported. This finding indicates the presence of a communication between the ILS and the airways, thus supporting the theory that ILSs are acquired lesions, rather than congenital malformations.
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19/19. Extralobar pulmonary sequestration with an associated cyst of mixed bronchogenic and esophageal type--a case report.

    We report an unusual case of extralobar pulmonary sequestration (ELS) with an associated cyst of mixed bronchogenic and esophageal type. A 58-year-old woman was incidentally found to have a 6 x 6 x 5 cm sized mass in the right superior mediastinum. The mass consisted of sequestrated pulmonary tissue and an unilocular cyst with a direct communication. The cyst could not be easily classified because it was lined by squamous or respiratory epithelium with two distinct muscle layers and bronchial glands. Bronchial cartilage was present in close proximity to the ELS. This unusual combination of ELS with a foregut cyst might be a part of bronchopulmonary foregut malformation, attributed to a common embryologic pathogenesis.
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