標題: Congenital atresia of unilateral pulmonary veins associated with a single ventricle - A rare case report and literature review
作者: Lee, Hsing-Yuan
Hwang, Betau
Lee, Pi-Chang
Jan, Sheng-Ling
Meng, C. C. Laura
生物科技學系
Department of Biological Science and Technology
關鍵字: congenital heart disease;pulmonary vein atresia;single ventricle
公開日期: 1-Sep-2008
摘要: Congenital atresia or extreme hypoplasia of individual pulmonary veins is a rare condition that is usually asymptomatic if it only involves 1 or 2 segments of the lungs. It may be fatal if it occurs in combination with other complex cardiac defects. The patients often present with recurrent pulmonary infections, hemoptysis, or cyanosis in the latter case. A definitive diagnosis can be made by cardiac catheterization with selective pulmonary wedge angiography. The treatment of this condition is a challenge and is controversial, and the prognosis is usually poor. We describe the case of a male infant who suffered from shortness of breath and cyanosis since birth. On echocardiography and cardiac catheterization, he was found to have a complex congenital heart disease with a single ventricle (right ventricle morphology), mitral atresia, large atrial septal defect, and atresia of the right pulmonary veins; severe pulmonary hypertension was also observed. He underwent pulmonary artery banding at the age of 2.5 months, but he died because of a pulmonary infection at the age of 6 months. In this report, we present this rare case and review the previous literature.
URI: http://dx.doi.org/10.1253/circj.CJ-07-0516
http://hdl.handle.net/11536/8380
ISSN: 1346-9843
DOI: 10.1253/circj.CJ-07-0516
期刊: CIRCULATION JOURNAL
Volume: 72
Issue: 9
起始頁: 1544
結束頁: 1546
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