Document Detail

Congenital atresia of unilateral pulmonary veins associated with a single ventricle: a rare case report and literature review.
MedLine Citation:
PMID:  18724038     Owner:  NLM     Status:  MEDLINE    
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.
Hsing-Yuan Lee; Betau Hwang; Pi-Chang Lee; Sheng-Ling Jan; C C Laura Meng
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Publication Detail:
Type:  Case Reports; Journal Article; Review    
Journal Detail:
Title:  Circulation journal : official journal of the Japanese Circulation Society     Volume:  72     ISSN:  1346-9843     ISO Abbreviation:  Circ. J.     Publication Date:  2008 Sep 
Date Detail:
Created Date:  2008-08-25     Completed Date:  2009-08-18     Revised Date:  -    
Medline Journal Info:
Nlm Unique ID:  101137683     Medline TA:  Circ J     Country:  Japan    
Other Details:
Languages:  eng     Pagination:  1544-6     Citation Subset:  IM    
Department of Pediatrics, Taoyuan Armed Force General Hospital, Taoyuan, Taiwan.
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MeSH Terms
Fatal Outcome
Heart Catheterization
Heart Ventricles / radiography*
Pneumonia / etiology,  radiography,  therapy
Pulmonary Atresia / complications,  radiography*,  therapy
Pulmonary Veins / radiography*

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