Lymphoscintigraphy in plastic bronchitis, A pediatric case report
Identifieur interne : 000790 ( Istex/Corpus ); précédent : 000789; suivant : 000791Lymphoscintigraphy in plastic bronchitis, A pediatric case report
Auteurs : Dima R. Ezmigna ; Wayne J. Morgan ; Marlys H. Witte ; Mark A. BrownSource :
- Pediatric Pulmonology [ 8755-6863 ] ; 2013-05.
Abstract
Plastic bronchitis (PB) is an uncommon, potentially fatal disease, marked by endobronchial cast formation causing variable degrees of respiratory distress. Primary and secondary pulmonary lymphatic abnormalities have been identified among the underlying mechanisms of cast formation. We present a case of PB where lymphoscintigraphy demonstrated the underlying lymphatic defect. A 6‐year‐old Hispanic male with congenital heart disease (CHD; post‐Fontan) presented with recurrent pneumonia, respiratory distress. Bronchoscopy showed inflamed hypervascular mucosa and thick mucus plugs; no casts were seen. Later, PB was diagnosed after the patient expectorated a bronchial cast. Cast analysis showed lymphocytic aggregates with mucin and fibrin. Lymphoscintigraphy revealed abnormal lymphatic collaterals and retrograde trace reflux into the superior mediastinum, a picture consistent with thoracic duct lymph leakage into the tracheobronchial tree. The pathogenesis of PB is not fully understood, especially in patients with CHD. Chyle in bronchial casts suggests abnormal lymphatic flow. Reports of lymph flow abnormalities, especially endobronchial lymph leakage in CHD are limited. Lymphoscintigraphy in our case demonstrated clear evidence of retrograde lymph reflux and leakage into the bronchial tree. The case presented suggests that in some patients following Fontan surgery, high intrathoracic lymphatic pressure and retrograde lymph flow may contribute to recurrent cast formation. Finding the underlying lymphatic abnormality helps in specific case management. Lymphoscintigraphy is a safer and easier method than lymphangiography. Surgical lymphatic–venous shunting may be possible in select cases. Pediatr Pulmonol. 2013; 48:515–518. © 2012 Wiley Periodicals, Inc.
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DOI: 10.1002/ppul.22673
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Ezmigna</name><affiliation><mods:affiliation>Department of Pediatrics, University of Arizona, Tucson, Arizona</mods:affiliation></affiliation><affiliation><mods:affiliation>Department of Pediatrics, University of Arizona, 1501 N Cambell Ave., Tucson, AZ 85724.</mods:affiliation></affiliation></author><author><name sortKey="Morgan, Wayne J" sort="Morgan, Wayne J" uniqKey="Morgan W" first="Wayne J." last="Morgan">Wayne J. Morgan</name><affiliation><mods:affiliation>Department of Pediatrics, University of Arizona, Tucson, Arizona</mods:affiliation></affiliation></author><author><name sortKey="Witte, Marlys H" sort="Witte, Marlys H" uniqKey="Witte M" first="Marlys H." last="Witte">Marlys H. Witte</name><affiliation><mods:affiliation>Department of Surgery, University of Arizona, Tucson, Arizona</mods:affiliation></affiliation></author><author><name sortKey="Brown, Mark A" sort="Brown, Mark A" uniqKey="Brown M" first="Mark A." last="Brown">Mark A. Brown</name><affiliation><mods:affiliation>Department of Pediatrics, University of Arizona, Tucson, Arizona</mods:affiliation></affiliation></author></analytic><monogr></monogr><series><title level="j" type="main">Pediatric Pulmonology</title><title level="j" type="alt">PEDIATRIC PULMONOLOGY</title><idno type="ISSN">8755-6863</idno><idno type="eISSN">1099-0496</idno><imprint><biblScope unit="vol">48</biblScope><biblScope unit="issue">5</biblScope><biblScope unit="page" from="515">515</biblScope><biblScope unit="page" to="518">518</biblScope><biblScope unit="page-count">4</biblScope><publisher>Wiley Subscription Services, Inc., A Wiley Company</publisher><pubPlace>Hoboken</pubPlace><date type="published" when="2013-05">2013-05</date></imprint><idno type="ISSN">8755-6863</idno></series></biblStruct></sourceDesc><seriesStmt><idno type="ISSN">8755-6863</idno></seriesStmt></fileDesc><profileDesc><textClass></textClass></profileDesc></teiHeader><front><div type="abstract" xml:lang="en">Plastic bronchitis (PB) is an uncommon, potentially fatal disease, marked by endobronchial cast formation causing variable degrees of respiratory distress. Primary and secondary pulmonary lymphatic abnormalities have been identified among the underlying mechanisms of cast formation. We present a case of PB where lymphoscintigraphy demonstrated the underlying lymphatic defect. A 6‐year‐old Hispanic male with congenital heart disease (CHD; post‐Fontan) presented with recurrent pneumonia, respiratory distress. Bronchoscopy showed inflamed hypervascular mucosa and thick mucus plugs; no casts were seen. Later, PB was diagnosed after the patient expectorated a bronchial cast. Cast analysis showed lymphocytic aggregates with mucin and fibrin. Lymphoscintigraphy revealed abnormal lymphatic collaterals and retrograde trace reflux into the superior mediastinum, a picture consistent with thoracic duct lymph leakage into the tracheobronchial tree. The pathogenesis of PB is not fully understood, especially in patients with CHD. Chyle in bronchial casts suggests abnormal lymphatic flow. Reports of lymph flow abnormalities, especially endobronchial lymph leakage in CHD are limited. Lymphoscintigraphy in our case demonstrated clear evidence of retrograde lymph reflux and leakage into the bronchial tree. The case presented suggests that in some patients following Fontan surgery, high intrathoracic lymphatic pressure and retrograde lymph flow may contribute to recurrent cast formation. Finding the underlying lymphatic abnormality helps in specific case management. Lymphoscintigraphy is a safer and easier method than lymphangiography. Surgical lymphatic–venous shunting may be possible in select cases. 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Primary and secondary pulmonary lymphatic abnormalities have been identified among the underlying mechanisms of cast formation. We present a case of PB where lymphoscintigraphy demonstrated the underlying lymphatic defect. A 6‐year‐old Hispanic male with congenital heart disease (CHD; post‐Fontan) presented with recurrent pneumonia, respiratory distress. Bronchoscopy showed inflamed hypervascular mucosa and thick mucus plugs; no casts were seen. Later, PB was diagnosed after the patient expectorated a bronchial cast. Cast analysis showed lymphocytic aggregates with mucin and fibrin. Lymphoscintigraphy revealed abnormal lymphatic collaterals and retrograde trace reflux into the superior mediastinum, a picture consistent with thoracic duct lymph leakage into the tracheobronchial tree. The pathogenesis of PB is not fully understood, especially in patients with CHD. Chyle in bronchial casts suggests abnormal lymphatic flow. Reports of lymph flow abnormalities, especially endobronchial lymph leakage in CHD are limited. Lymphoscintigraphy in our case demonstrated clear evidence of retrograde lymph reflux and leakage into the bronchial tree. The case presented suggests that in some patients following Fontan surgery, high intrathoracic lymphatic pressure and retrograde lymph flow may contribute to recurrent cast formation. Finding the underlying lymphatic abnormality helps in specific case management. Lymphoscintigraphy is a safer and easier method than lymphangiography. Surgical lymphatic–venous shunting may be possible in select cases. 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role="corresp"><persName><forename type="first">Dima R.</forename><surname>Ezmigna</surname><roleName type="degree">MD</roleName></persName><email>dezmigna@yahoo.com</email><affiliation>Department of Pediatrics, University of Arizona, Tucson, Arizona<address><country key="US"></country></address></affiliation><affiliation>Department of Pediatrics, University of Arizona, 1501 N Cambell Ave., Tucson, AZ 85724.</affiliation></author><author xml:id="author-0001"><persName><forename type="first">Wayne J.</forename><surname>Morgan</surname><roleName type="degree">MD</roleName></persName><affiliation>Department of Pediatrics, University of Arizona, Tucson, Arizona<address><country key="US"></country></address></affiliation></author><author xml:id="author-0002"><persName><forename type="first">Marlys H.</forename><surname>Witte</surname><roleName type="degree">MD</roleName></persName><affiliation>Department of Surgery, University of Arizona, Tucson, Arizona<address><country 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Primary and secondary pulmonary lymphatic abnormalities have been identified among the underlying mechanisms of cast formation. We present a case of PB where lymphoscintigraphy demonstrated the underlying lymphatic defect. A 6‐year‐old Hispanic male with congenital heart disease (CHD; post‐Fontan) presented with recurrent pneumonia, respiratory distress. Bronchoscopy showed inflamed hypervascular mucosa and thick mucus plugs; no casts were seen. Later, PB was diagnosed after the patient expectorated a bronchial cast. Cast analysis showed lymphocytic aggregates with mucin and fibrin. Lymphoscintigraphy revealed abnormal lymphatic collaterals and retrograde trace reflux into the superior mediastinum, a picture consistent with thoracic duct lymph leakage into the tracheobronchial tree. The pathogenesis of PB is not fully understood, especially in patients with CHD. Chyle in bronchial casts suggests abnormal lymphatic flow. Reports of lymph flow abnormalities, especially endobronchial lymph leakage in CHD are limited. Lymphoscintigraphy in our case demonstrated clear evidence of retrograde lymph reflux and leakage into the bronchial tree. The case presented suggests that in some patients following Fontan surgery, high intrathoracic lymphatic pressure and retrograde lymph flow may contribute to recurrent cast formation. Finding the underlying lymphatic abnormality helps in specific case management. Lymphoscintigraphy is a safer and easier method than lymphangiography. Surgical lymphatic–venous shunting may be possible in select cases. Pediatr Pulmonol. 2013; 48:515–518. © 2012 Wiley Periodicals, Inc.</p></abstract><textClass><keywords xml:lang="en"><term xml:id="kwd1">plastic bronchitis</term><term xml:id="kwd2">lymphoscintigraphy</term><term xml:id="kwd3">congenital heart disease</term><term xml:id="kwd4">lymph leakage</term></keywords><classCode scheme="articleCategory">Case Report</classCode><classCode scheme="tocHeading1">Case Reports</classCode></textClass><langUsage><language ident="EN"></language></langUsage></profileDesc></teiHeader></istex:fulltextTEI><json:item><extension>txt</extension><original>false</original><mimetype>text/plain</mimetype><uri>https://api.istex.fr/document/111680058EB0D7FC180A5B98038F12E503537B62/fulltext/txt</uri></json:item></fulltext><metadata><istex:metadataXml wicri:clean="Wiley, elements deleted: body"><istex:xmlDeclaration>version="1.0" encoding="UTF-8" standalone="yes"</istex:xmlDeclaration><istex:document><component version="2.0" type="serialArticle" xml:lang="en"><header><publicationMeta level="product"><publisherInfo><publisherName>Wiley Subscription Services, Inc., A Wiley Company</publisherName><publisherLoc>Hoboken</publisherLoc></publisherInfo><doi registered="yes">10.1002/(ISSN)1099-0496</doi><issn type="print">8755-6863</issn><issn type="electronic">1099-0496</issn><idGroup><id type="product" value="PPUL"></id></idGroup><titleGroup><title type="main" xml:lang="en" sort="PEDIATRIC PULMONOLOGY">Pediatric Pulmonology</title><title type="short">Pediatr. 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Primary and secondary pulmonary lymphatic abnormalities have been identified among the underlying mechanisms of cast formation. We present a case of PB where lymphoscintigraphy demonstrated the underlying lymphatic defect. A 6‐year‐old Hispanic male with congenital heart disease (CHD; post‐Fontan) presented with recurrent pneumonia, respiratory distress. Bronchoscopy showed inflamed hypervascular mucosa and thick mucus plugs; no casts were seen. Later, PB was diagnosed after the patient expectorated a bronchial cast. Cast analysis showed lymphocytic aggregates with mucin and fibrin. Lymphoscintigraphy revealed abnormal lymphatic collaterals and retrograde trace reflux into the superior mediastinum, a picture consistent with thoracic duct lymph leakage into the tracheobronchial tree. The pathogenesis of PB is not fully understood, especially in patients with CHD. Chyle in bronchial casts suggests abnormal lymphatic flow. Reports of lymph flow abnormalities, especially endobronchial lymph leakage in CHD are limited. Lymphoscintigraphy in our case demonstrated clear evidence of retrograde lymph reflux and leakage into the bronchial tree. The case presented suggests that in some patients following Fontan surgery, high intrathoracic lymphatic pressure and retrograde lymph flow may contribute to recurrent cast formation. Finding the underlying lymphatic abnormality helps in specific case management. Lymphoscintigraphy is a safer and easier method than lymphangiography. Surgical lymphatic–venous shunting may be possible in select cases. Pediatr Pulmonol. 2013; 48:515–518. © 2012 Wiley Periodicals, Inc.</p></abstract></abstractGroup></contentMeta><noteGroup><note xml:id="fn1"><p>Marlys Witte and Mark Brown contributed equally to this work.</p></note><note xml:id="fn2"><p>Conflict of interest: None.</p></note></noteGroup></header></component></istex:document></istex:metadataXml><mods version="3.6"><titleInfo lang="en"><title>Lymphoscintigraphy in plastic bronchitis, A pediatric case report</title></titleInfo><titleInfo type="abbreviated" lang="en"><title>Lymphoscintigraphy In Plastic Bronchitis</title></titleInfo><titleInfo type="alternative" contentType="CDATA" lang="en"><title>Lymphoscintigraphy in plastic bronchitis, A pediatric case report</title></titleInfo><name type="personal"><namePart type="given">Dima R.</namePart><namePart type="family">Ezmigna</namePart><namePart type="termsOfAddress">MD</namePart><affiliation>Department of Pediatrics, University of Arizona, Tucson, Arizona</affiliation><affiliation>Department of Pediatrics, University of Arizona, 1501 N Cambell Ave., Tucson, AZ 85724.</affiliation><role><roleTerm type="text">author</roleTerm></role></name><name type="personal"><namePart type="given">Wayne J.</namePart><namePart type="family">Morgan</namePart><namePart type="termsOfAddress">MD</namePart><affiliation>Department of Pediatrics, University of Arizona, Tucson, Arizona</affiliation><role><roleTerm type="text">author</roleTerm></role></name><name type="personal"><namePart type="given">Marlys H.</namePart><namePart type="family">Witte</namePart><namePart type="termsOfAddress">MD</namePart><affiliation>Department of Surgery, University of Arizona, Tucson, Arizona</affiliation><role><roleTerm type="text">author</roleTerm></role></name><name type="personal"><namePart type="given">Mark A.</namePart><namePart type="family">Brown</namePart><namePart type="termsOfAddress">MD</namePart><affiliation>Department of Pediatrics, University of Arizona, Tucson, Arizona</affiliation><role><roleTerm type="text">author</roleTerm></role></name><typeOfResource>text</typeOfResource><genre type="case-report" displayLabel="caseStudy"></genre><originInfo><publisher>Wiley Subscription Services, Inc., A Wiley Company</publisher><place><placeTerm type="text">Hoboken</placeTerm></place><dateIssued encoding="w3cdtf">2013-05</dateIssued><dateCaptured encoding="w3cdtf">2012-03-01</dateCaptured><dateValid encoding="w3cdtf">2012-06-18</dateValid><copyrightDate encoding="w3cdtf">2013</copyrightDate></originInfo><language><languageTerm type="code" authority="rfc3066">en</languageTerm><languageTerm type="code" authority="iso639-2b">eng</languageTerm></language><physicalDescription><internetMediaType>text/html</internetMediaType><extent unit="figures">2</extent><extent unit="references">15</extent><extent unit="words">2615</extent></physicalDescription><abstract lang="en">Plastic bronchitis (PB) is an uncommon, potentially fatal disease, marked by endobronchial cast formation causing variable degrees of respiratory distress. Primary and secondary pulmonary lymphatic abnormalities have been identified among the underlying mechanisms of cast formation. We present a case of PB where lymphoscintigraphy demonstrated the underlying lymphatic defect. A 6‐year‐old Hispanic male with congenital heart disease (CHD; post‐Fontan) presented with recurrent pneumonia, respiratory distress. Bronchoscopy showed inflamed hypervascular mucosa and thick mucus plugs; no casts were seen. Later, PB was diagnosed after the patient expectorated a bronchial cast. Cast analysis showed lymphocytic aggregates with mucin and fibrin. Lymphoscintigraphy revealed abnormal lymphatic collaterals and retrograde trace reflux into the superior mediastinum, a picture consistent with thoracic duct lymph leakage into the tracheobronchial tree. The pathogenesis of PB is not fully understood, especially in patients with CHD. Chyle in bronchial casts suggests abnormal lymphatic flow. Reports of lymph flow abnormalities, especially endobronchial lymph leakage in CHD are limited. Lymphoscintigraphy in our case demonstrated clear evidence of retrograde lymph reflux and leakage into the bronchial tree. The case presented suggests that in some patients following Fontan surgery, high intrathoracic lymphatic pressure and retrograde lymph flow may contribute to recurrent cast formation. Finding the underlying lymphatic abnormality helps in specific case management. Lymphoscintigraphy is a safer and easier method than lymphangiography. Surgical lymphatic–venous shunting may be possible in select cases. Pediatr Pulmonol. 2013; 48:515–518. © 2012 Wiley Periodicals, Inc.</abstract><note type="content">*Marlys Witte and Mark Brown contributed equally to this work.</note><note type="content">*Conflict of interest: None.</note><note type="funding">University of Arizona</note><subject lang="en"><genre>keywords</genre><topic>plastic bronchitis</topic><topic>lymphoscintigraphy</topic><topic>congenital heart disease</topic><topic>lymph leakage</topic></subject><relatedItem type="host"><titleInfo><title>Pediatric Pulmonology</title></titleInfo><titleInfo type="abbreviated"><title>Pediatr. Pulmonol.</title></titleInfo><genre type="journal">journal</genre><subject><genre>article-category</genre><topic>Case Report</topic></subject><identifier type="ISSN">8755-6863</identifier><identifier type="eISSN">1099-0496</identifier><identifier type="DOI">10.1002/(ISSN)1099-0496</identifier><identifier type="PublisherID">PPUL</identifier><part><date>2013</date><detail type="volume"><caption>vol.</caption><number>48</number></detail><detail type="issue"><caption>no.</caption><number>5</number></detail><extent unit="pages"><start>515</start><end>518</end><total>4</total></extent></part></relatedItem><identifier type="istex">111680058EB0D7FC180A5B98038F12E503537B62</identifier><identifier type="DOI">10.1002/ppul.22673</identifier><identifier type="ArticleID">PPUL22673</identifier><accessCondition type="use and reproduction" contentType="copyright">Copyright © 2012 Wiley Periodicals, Inc.</accessCondition><recordInfo><recordContentSource>WILEY</recordContentSource><recordOrigin>Wiley Subscription Services, Inc., A Wiley Company</recordOrigin></recordInfo></mods></metadata><serie></serie></istex></record>Pour manipuler ce document sous Unix (Dilib)
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