Hyperlucent lung secondary to homocystinuria
Identifieur interne : 002A92 ( Main/Merge ); précédent : 002A91; suivant : 002A93Hyperlucent lung secondary to homocystinuria
Auteurs : J. Herman [États-Unis] ; J. H. Miller [États-Unis] ; F. Wang [États-Unis]Source :
- Pediatric Radiology [ 0301-0449 ] ; 1996-09-01.
English descriptors
- Teeft :
- Abnormality, Bronchiolitis obliterans, Cystathionine, Homocystine, Homocystinuria, Hyperlucent, Hyperlucent lung, Irvine, Lung fields, Lung scanning, Nephrotic syndrome, Pediatr radiol, Perfusion, Perfusion scintiphoto, Pulmonary arteries, Right lung, Right side, Skeletal abnormalities, Syndrome, Thrombosis, Uniform ventilation.
Abstract
Abstract: At 23 months of age, one of a pair of monozygotic twins with radiographic unilateral hyperlucent lung was evaluated by radionuclide ventilation/perfusion pulmonary studies, which revealed a ventilation/perfusion mismatch of an entire lung. This twin died, and autopsy revealed pulmonary arterial thrombosis and histological changes compatible with homocystinuria, which was subsequently shown to be present in the surviving twin as well. A ventilation/perfusion lung scan of the surviving twin revealed multiple ventilation/perfusion mismatched defects, suggestive of pulmonary embolism. The presenting manifestation of homocystinuria in these patients was the pulmonary thrombotic disease. Neither twin had any other stigmata of homocystinuria at the time of initial presentation.
Url:
DOI: 10.1007/BF01356832
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- to stream Istex, to step Curation: 001762
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ISTEX:769EF9E6E3F7D9E3EC4EC808FE37D3BBC1294405Le document en format XML
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<author><name sortKey="Miller, J H" sort="Miller, J H" uniqKey="Miller J" first="J. H." last="Miller">J. H. Miller</name>
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<author><name sortKey="Wang, F" sort="Wang, F" uniqKey="Wang F" first="F." last="Wang">F. Wang</name>
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<author><name sortKey="Miller, J H" sort="Miller, J H" uniqKey="Miller J" first="J. H." last="Miller">J. H. Miller</name>
<affiliation wicri:level="2"><country xml:lang="fr">États-Unis</country>
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<author><name sortKey="Wang, F" sort="Wang, F" uniqKey="Wang F" first="F." last="Wang">F. Wang</name>
<affiliation wicri:level="2"><country xml:lang="fr">États-Unis</country>
<wicri:regionArea>Department of Radiology, University of California, Irvine Medical Center, Irvine, California</wicri:regionArea>
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<series><title level="j">Pediatric Radiology</title>
<title level="j" type="sub">Roentgenology, Nuclear Medicine, Ultrasound, CT, MRI</title>
<title level="j" type="abbrev">Pediatr Radiol</title>
<idno type="ISSN">0301-0449</idno>
<idno type="eISSN">1432-1998</idno>
<imprint><publisher>Springer-Verlag</publisher>
<pubPlace>Berlin/Heidelberg</pubPlace>
<date type="published" when="1996-09-01">1996-09-01</date>
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<profileDesc><textClass><keywords scheme="Teeft" xml:lang="en"><term>Abnormality</term>
<term>Bronchiolitis obliterans</term>
<term>Cystathionine</term>
<term>Homocystine</term>
<term>Homocystinuria</term>
<term>Hyperlucent</term>
<term>Hyperlucent lung</term>
<term>Irvine</term>
<term>Lung fields</term>
<term>Lung scanning</term>
<term>Nephrotic syndrome</term>
<term>Pediatr radiol</term>
<term>Perfusion</term>
<term>Perfusion scintiphoto</term>
<term>Pulmonary arteries</term>
<term>Right lung</term>
<term>Right side</term>
<term>Skeletal abnormalities</term>
<term>Syndrome</term>
<term>Thrombosis</term>
<term>Uniform ventilation</term>
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<front><div type="abstract" xml:lang="en">Abstract: At 23 months of age, one of a pair of monozygotic twins with radiographic unilateral hyperlucent lung was evaluated by radionuclide ventilation/perfusion pulmonary studies, which revealed a ventilation/perfusion mismatch of an entire lung. This twin died, and autopsy revealed pulmonary arterial thrombosis and histological changes compatible with homocystinuria, which was subsequently shown to be present in the surviving twin as well. A ventilation/perfusion lung scan of the surviving twin revealed multiple ventilation/perfusion mismatched defects, suggestive of pulmonary embolism. The presenting manifestation of homocystinuria in these patients was the pulmonary thrombotic disease. Neither twin had any other stigmata of homocystinuria at the time of initial presentation.</div>
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