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Multimodality lymphatic imaging of postoperative chylothorax in an infant with Noonan syndrome: a case report

BACKGROUND: Chylothorax is a rare complication of pediatric cardiac operations that occurs more frequently in children with Noonan syndrome, a genetic disorder associated with cardiac defects and lymphatic anomalies. CASE PRESENTATION: We report a case of postoperative chylothorax in a 6-month-old i...

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Autores principales: Pham, Kay T., Balaguru, Duraisamy, Tammisetti, Varaha S., Guevara, Carlos J., Rasmussen, John C., Zvavanjanja, Rodrick C., Hanfland, Robert, Sevick-Muraca, Eva M., Aldrich, Melissa B.
Formato: Online Artículo Texto
Lenguaje:English
Publicado: BioMed Central 2020
Materias:
Acceso en línea:https://www.ncbi.nlm.nih.gov/pmc/articles/PMC7640692/
https://www.ncbi.nlm.nih.gov/pubmed/33148331
http://dx.doi.org/10.1186/s40001-020-00455-w
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author Pham, Kay T.
Balaguru, Duraisamy
Tammisetti, Varaha S.
Guevara, Carlos J.
Rasmussen, John C.
Zvavanjanja, Rodrick C.
Hanfland, Robert
Sevick-Muraca, Eva M.
Aldrich, Melissa B.
author_facet Pham, Kay T.
Balaguru, Duraisamy
Tammisetti, Varaha S.
Guevara, Carlos J.
Rasmussen, John C.
Zvavanjanja, Rodrick C.
Hanfland, Robert
Sevick-Muraca, Eva M.
Aldrich, Melissa B.
author_sort Pham, Kay T.
collection PubMed
description BACKGROUND: Chylothorax is a rare complication of pediatric cardiac operations that occurs more frequently in children with Noonan syndrome, a genetic disorder associated with cardiac defects and lymphatic anomalies. CASE PRESENTATION: We report a case of postoperative chylothorax in a 6-month-old infant with Noonan syndrome where multimodality lymphatic imaging guided management was followed. Drainage patterns of the lymphatic capillaries in the lower and upper extremities were visualized during near-infrared fluorescence lymphatic imaging (NIRFLI). Dynamic magnetic resonance lymphangiography (MRL) further identified the site of leakage in the thoracic duct and subsequently guided surgical intervention. CONCLUSIONS: Application of multimodality imaging allows for greater individualization of treatment and should be considered in patients with complex cases such as those with syndromes associated with a higher incidence of chylothorax. IRB Number: HSC-MS-13–0754, December 10, 2013
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spelling pubmed-76406922020-11-05 Multimodality lymphatic imaging of postoperative chylothorax in an infant with Noonan syndrome: a case report Pham, Kay T. Balaguru, Duraisamy Tammisetti, Varaha S. Guevara, Carlos J. Rasmussen, John C. Zvavanjanja, Rodrick C. Hanfland, Robert Sevick-Muraca, Eva M. Aldrich, Melissa B. Eur J Med Res Case Report BACKGROUND: Chylothorax is a rare complication of pediatric cardiac operations that occurs more frequently in children with Noonan syndrome, a genetic disorder associated with cardiac defects and lymphatic anomalies. CASE PRESENTATION: We report a case of postoperative chylothorax in a 6-month-old infant with Noonan syndrome where multimodality lymphatic imaging guided management was followed. Drainage patterns of the lymphatic capillaries in the lower and upper extremities were visualized during near-infrared fluorescence lymphatic imaging (NIRFLI). Dynamic magnetic resonance lymphangiography (MRL) further identified the site of leakage in the thoracic duct and subsequently guided surgical intervention. CONCLUSIONS: Application of multimodality imaging allows for greater individualization of treatment and should be considered in patients with complex cases such as those with syndromes associated with a higher incidence of chylothorax. IRB Number: HSC-MS-13–0754, December 10, 2013 BioMed Central 2020-11-04 /pmc/articles/PMC7640692/ /pubmed/33148331 http://dx.doi.org/10.1186/s40001-020-00455-w Text en © The Author(s) 2020 Open AccessThis article is licensed under a Creative Commons Attribution 4.0 International License, which permits use, sharing, adaptation, distribution and reproduction in any medium or format, as long as you give appropriate credit to the original author(s) and the source, provide a link to the Creative Commons licence, and indicate if changes were made. The images or other third party material in this article are included in the article's Creative Commons licence, unless indicated otherwise in a credit line to the material. If material is not included in the article's Creative Commons licence and your intended use is not permitted by statutory regulation or exceeds the permitted use, you will need to obtain permission directly from the copyright holder. To view a copy of this licence, visit http://creativecommons.org/licenses/by/4.0/. The Creative Commons Public Domain Dedication waiver (http://creativecommons.org/publicdomain/zero/1.0/) applies to the data made available in this article, unless otherwise stated in a credit line to the data.
spellingShingle Case Report
Pham, Kay T.
Balaguru, Duraisamy
Tammisetti, Varaha S.
Guevara, Carlos J.
Rasmussen, John C.
Zvavanjanja, Rodrick C.
Hanfland, Robert
Sevick-Muraca, Eva M.
Aldrich, Melissa B.
Multimodality lymphatic imaging of postoperative chylothorax in an infant with Noonan syndrome: a case report
title Multimodality lymphatic imaging of postoperative chylothorax in an infant with Noonan syndrome: a case report
title_full Multimodality lymphatic imaging of postoperative chylothorax in an infant with Noonan syndrome: a case report
title_fullStr Multimodality lymphatic imaging of postoperative chylothorax in an infant with Noonan syndrome: a case report
title_full_unstemmed Multimodality lymphatic imaging of postoperative chylothorax in an infant with Noonan syndrome: a case report
title_short Multimodality lymphatic imaging of postoperative chylothorax in an infant with Noonan syndrome: a case report
title_sort multimodality lymphatic imaging of postoperative chylothorax in an infant with noonan syndrome: a case report
topic Case Report
url https://www.ncbi.nlm.nih.gov/pmc/articles/PMC7640692/
https://www.ncbi.nlm.nih.gov/pubmed/33148331
http://dx.doi.org/10.1186/s40001-020-00455-w
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