PubMed · 13350032
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E SCHIEPPATI, E MEILIJ, A H VILLEGAS. 1956-03-23. [Sequestration pulmonary lobectomy].. https://pubmed.ncbi.nlm.nih.gov/13350032/
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Bronchopulmonary sequestration is a rare malformation. Is characterized by non-functioning lung tissue fed from one or several aberrant systemic arteries. Authors present a 5-month-old boy with Hirschsprung disease in whom intralobar sequestration was fortuitously made on the occasion of pneumopathy. Sequestration with associated Hirschsprung disease is exceptional and not reported in the literature.
Extralobar pulmonary sequestration (ELS) and congenital cystic adenomatoid malformation have been reported to coexist in several variations. This suggests a common embryologic origin. A 6-month-old boy presented with a history of recurrent pneumonias. The patient was diagnosed with a right lower lobe congenital cystic adenomatoid malformation (CCAM) and a left lower lobe ELS/CCAM. The diagnosis was made with the aid of a multidetector computed tomography (CT). Three-dimensional CT reconstruction showed the presence of a right lower lobe CCAM, a left lower ELS with an aberrant arterial supply from the celiac axis, and possible venous drainage into the right CCAM. The patient underwent a right thoracotomy. Intraoperatively, the lesions were discovered to be connected by a band of tissue. The right lower lobe CCAM and the left ELS were removed from the right chest. Histologic analysis confirmed the presence of a CCAM within the right lower lobe. The ELS had involvement of a type II CCAM within the sequestration. The connection between the right CCAM and left ELS/CCAM showed an anomalous conducting airway, anomalous vein, and anomalous artery connecting the 2 lesions. The authors present the first case of a CCAM connected to an ELS/CCAM in the contralateral hemithorax. The unique anatomic configuration of these lesions suggests a common embryologic origin of ELS and CCAM.