Practical Considerations for Endovascular Treatment of Pulmonary Sequestration-Induced Hemoptysis: Two Case Reports and Review of Literature

Abstract

Pulmonary sequestration is a rare condition in which a segment or lobe of dysplastic lung tissue develops without connecting to the airways and pulmonary arteries. Hemoptysis is a potentially fatal complication of pulmonary sequestration. The current report discusses two complex cases of pulmonary sequestration. The first involves a patient with coexisting partial anomalous pulmonary venous drainage, in which hemoptysis was effectively managed through embolization of the abnormal systemic artery. Embolization of the anomalous pulmonary vein was not performed because the left-to-right shunt was hemodynamically insignificant. The second case involved a patient with a multi-cystic lung mass and air-fluid levels. Although embolization of the abnormal systemic artery controls hemoptysis, it also leads to prolonged complications due to aggravated inflammation. When providing endovascular treatment for pulmonary sequestration, conducting a comprehensive evaluation using pretreatment imaging is necessary to tailor the approach to each patient's unique condition.

Keywords: Bronchopulmonary Sequestration; Lung Abscess; Scimitar Syndrome; Therapeutic Embolization; ‌Hemoptysis.

Fig. 1. Case findings of a 32-year-old male patient with pulmonary sequestration.

A, B. Contrast-enhanced chest CT scan shows tortuous anomalous pulmonary vein (asterisks) draining into the inferior vena cava and an abnormal systemic feeding artery (arrow) from descending thoracic aorta. Focal ground glass opacity (arrowhead) is also noted in the right lower lobe. C. The volume-rendered image demonstrates dual venous drainage into the right superior pulmonary vein (arrowhead) and inferior vena cava (asterisks). No normal right lower pulmonary vein is visible. D, E. Digital subtraction angiography confirms abnormal systemic artery (arrow) and anomalous pulmonary veins (arrowhead, asterisks). F. Post-embolization CT confirms glue cast (arrowheads) in the abnormal systemic artery.

Fig. 2. Case findings of a 33-year-old male patient with pulmonary sequestration.

A. Chest radiography reveals multiple cystic lesions with air-fluid levels (arrows) in the right lower lung. B, C. Contrast-enhanced chest CT scan demonstrates thin septations with air-fluid levels (asterisks) in multi-cystic lesions. An abnormal vascular structure (arrow) from the celiac axis is also visible. D. Digital subtraction angiography of the celiac axis confirms abnormal systemic artery (arrow) supplying multi-cystic lesions. E. After embolization, a glue cast is noted in the abnormal systemic artery. F. Follow-up chest CT scan reveals a marked increase of fluid (arrows) in the multi-cystic lesion. Glue cast (arrowhead) is also noted in the abnormal systemic artery.