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Percutaneous Lymphatic Embolization of Abnormal Pulmonary Lymphatic Flow as Treatment of Plastic Bronchitis in Patients With Congenital Heart Disease

BACKGROUND—Plastic bronchitis is a potentially fatal disorder occurring in children with single-ventricle physiology, and other diseases, as well, such as asthma. In this study, we report findings of abnormal pulmonary lymphatic flow, demonstrated by MRI lymphatic imaging, in patients with plastic b...

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Published in:Circulation (New York, N.Y.) N.Y.), 2016-03, Vol.133 (12), p.1160-1170
Main Authors: Dori, Yoav, Keller, Marc S, Rome, Jonathan J, Gillespie, Matthew J, Glatz, Andrew C, Dodds, Kathryn, Goldberg, David J, Goldfarb, Samuel, Rychik, Jack, Itkin, Maxim
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container_title Circulation (New York, N.Y.)
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creator Dori, Yoav
Keller, Marc S
Rome, Jonathan J
Gillespie, Matthew J
Glatz, Andrew C
Dodds, Kathryn
Goldberg, David J
Goldfarb, Samuel
Rychik, Jack
Itkin, Maxim
description BACKGROUND—Plastic bronchitis is a potentially fatal disorder occurring in children with single-ventricle physiology, and other diseases, as well, such as asthma. In this study, we report findings of abnormal pulmonary lymphatic flow, demonstrated by MRI lymphatic imaging, in patients with plastic bronchitis and percutaneous lymphatic intervention as a treatment for these patients. METHODS AND RESULTS—This is a retrospective case series of 18 patients with surgically corrected congenital heart disease and plastic bronchitis who presented for lymphatic imaging and intervention. Lymphatic imaging included heavy T2-weighted MRI and dynamic contrast-enhanced magnetic resonance lymphangiogram. All patients underwent bilateral intranodal lymphangiogram, and most patients underwent percutaneous lymphatic intervention. In 16 of 18 patients, MRI or lymphangiogram or both demonstrated retrograde lymphatic flow from the thoracic duct toward lung parenchyma. Intranodal lymphangiogram and thoracic duct catheterization was successful in all patients. Seventeen of 18 patients underwent either lymphatic embolization procedures or thoracic duct stenting with covered stents to exclude retrograde flow into the lungs. One of the 2 patients who did not have retrograde lymphatic flow did not undergo a lymphatic interventional procedure. A total of 15 of 17(88%) patients who underwent an intervention had significant symptomatic improvement at a median follow-up of 315 days (range, 45–770 days). The most common complication observed was nonspecific transient abdominal pain and transient hypotension. CONCLUSIONS—In this study, we demonstrated abnormal pulmonary lymphatic perfusion in most patients with plastic bronchitis. Interruption of the lymphatic flow resulted in significant improvement of symptoms in these patients and, in some cases, at least temporary resolution of cast formation.
doi_str_mv 10.1161/CIRCULATIONAHA.115.019710
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In this study, we report findings of abnormal pulmonary lymphatic flow, demonstrated by MRI lymphatic imaging, in patients with plastic bronchitis and percutaneous lymphatic intervention as a treatment for these patients. METHODS AND RESULTS—This is a retrospective case series of 18 patients with surgically corrected congenital heart disease and plastic bronchitis who presented for lymphatic imaging and intervention. Lymphatic imaging included heavy T2-weighted MRI and dynamic contrast-enhanced magnetic resonance lymphangiogram. All patients underwent bilateral intranodal lymphangiogram, and most patients underwent percutaneous lymphatic intervention. In 16 of 18 patients, MRI or lymphangiogram or both demonstrated retrograde lymphatic flow from the thoracic duct toward lung parenchyma. Intranodal lymphangiogram and thoracic duct catheterization was successful in all patients. Seventeen of 18 patients underwent either lymphatic embolization procedures or thoracic duct stenting with covered stents to exclude retrograde flow into the lungs. One of the 2 patients who did not have retrograde lymphatic flow did not undergo a lymphatic interventional procedure. A total of 15 of 17(88%) patients who underwent an intervention had significant symptomatic improvement at a median follow-up of 315 days (range, 45–770 days). The most common complication observed was nonspecific transient abdominal pain and transient hypotension. CONCLUSIONS—In this study, we demonstrated abnormal pulmonary lymphatic perfusion in most patients with plastic bronchitis. Interruption of the lymphatic flow resulted in significant improvement of symptoms in these patients and, in some cases, at least temporary resolution of cast formation.</description><identifier>ISSN: 0009-7322</identifier><identifier>EISSN: 1524-4539</identifier><identifier>DOI: 10.1161/CIRCULATIONAHA.115.019710</identifier><identifier>PMID: 26864093</identifier><language>eng</language><publisher>United States: by the American College of Cardiology Foundation and the American Heart Association, Inc</publisher><subject>Adolescent ; Balloon Occlusion ; Bronchitis, Chronic - etiology ; Bronchitis, Chronic - therapy ; Bronchoscopy ; Cardiac Catheterization ; Cardiac Surgical Procedures - adverse effects ; Child ; Child, Preschool ; Embolization, Therapeutic - methods ; Ethiodized Oil - administration &amp; dosage ; Ethiodized Oil - therapeutic use ; Female ; Fontan Procedure ; Heart Bypass, Right - adverse effects ; Heart Defects, Congenital - surgery ; Heart Transplantation ; Humans ; Lymphatic Vessels - physiopathology ; Lymphography ; Magnetic Resonance Imaging ; Male ; Postoperative Complications - etiology ; Postoperative Complications - therapy ; Retrospective Studies</subject><ispartof>Circulation (New York, N.Y.), 2016-03, Vol.133 (12), p.1160-1170</ispartof><rights>2016 by the American College of Cardiology Foundation and the American Heart Association, Inc.</rights><rights>2016 American Heart Association, Inc.</rights><lds50>peer_reviewed</lds50><oa>free_for_read</oa><woscitedreferencessubscribed>false</woscitedreferencessubscribed><citedby>FETCH-LOGICAL-c4220-e0d84ef3d15fdf5dcdc017a93e5cccbe40fad9bc70623e400dc77136292a7aeb3</citedby><cites>FETCH-LOGICAL-c4220-e0d84ef3d15fdf5dcdc017a93e5cccbe40fad9bc70623e400dc77136292a7aeb3</cites></display><links><openurl>$$Topenurl_article</openurl><openurlfulltext>$$Topenurlfull_article</openurlfulltext><thumbnail>$$Tsyndetics_thumb_exl</thumbnail><link.rule.ids>314,780,784,27924,27925</link.rule.ids><backlink>$$Uhttps://www.ncbi.nlm.nih.gov/pubmed/26864093$$D View this record in MEDLINE/PubMed$$Hfree_for_read</backlink></links><search><creatorcontrib>Dori, Yoav</creatorcontrib><creatorcontrib>Keller, Marc S</creatorcontrib><creatorcontrib>Rome, Jonathan J</creatorcontrib><creatorcontrib>Gillespie, Matthew J</creatorcontrib><creatorcontrib>Glatz, Andrew C</creatorcontrib><creatorcontrib>Dodds, Kathryn</creatorcontrib><creatorcontrib>Goldberg, David J</creatorcontrib><creatorcontrib>Goldfarb, Samuel</creatorcontrib><creatorcontrib>Rychik, Jack</creatorcontrib><creatorcontrib>Itkin, Maxim</creatorcontrib><title>Percutaneous Lymphatic Embolization of Abnormal Pulmonary Lymphatic Flow as Treatment of Plastic Bronchitis in Patients With Congenital Heart Disease</title><title>Circulation (New York, N.Y.)</title><addtitle>Circulation</addtitle><description>BACKGROUND—Plastic bronchitis is a potentially fatal disorder occurring in children with single-ventricle physiology, and other diseases, as well, such as asthma. 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Seventeen of 18 patients underwent either lymphatic embolization procedures or thoracic duct stenting with covered stents to exclude retrograde flow into the lungs. One of the 2 patients who did not have retrograde lymphatic flow did not undergo a lymphatic interventional procedure. A total of 15 of 17(88%) patients who underwent an intervention had significant symptomatic improvement at a median follow-up of 315 days (range, 45–770 days). The most common complication observed was nonspecific transient abdominal pain and transient hypotension. CONCLUSIONS—In this study, we demonstrated abnormal pulmonary lymphatic perfusion in most patients with plastic bronchitis. 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Seventeen of 18 patients underwent either lymphatic embolization procedures or thoracic duct stenting with covered stents to exclude retrograde flow into the lungs. One of the 2 patients who did not have retrograde lymphatic flow did not undergo a lymphatic interventional procedure. A total of 15 of 17(88%) patients who underwent an intervention had significant symptomatic improvement at a median follow-up of 315 days (range, 45–770 days). The most common complication observed was nonspecific transient abdominal pain and transient hypotension. CONCLUSIONS—In this study, we demonstrated abnormal pulmonary lymphatic perfusion in most patients with plastic bronchitis. 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subjects Adolescent
Balloon Occlusion
Bronchitis, Chronic - etiology
Bronchitis, Chronic - therapy
Bronchoscopy
Cardiac Catheterization
Cardiac Surgical Procedures - adverse effects
Child
Child, Preschool
Embolization, Therapeutic - methods
Ethiodized Oil - administration & dosage
Ethiodized Oil - therapeutic use
Female
Fontan Procedure
Heart Bypass, Right - adverse effects
Heart Defects, Congenital - surgery
Heart Transplantation
Humans
Lymphatic Vessels - physiopathology
Lymphography
Magnetic Resonance Imaging
Male
Postoperative Complications - etiology
Postoperative Complications - therapy
Retrospective Studies
title Percutaneous Lymphatic Embolization of Abnormal Pulmonary Lymphatic Flow as Treatment of Plastic Bronchitis in Patients With Congenital Heart Disease
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