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A Case of Suspected Adverse Reactions to Sirolimus in the Treatment of Generalized Lymphatic Anomaly

Generalized lymphatic anomaly (GLA) is characterized by diffuse or multicentric proliferation of dilated lymphatic vessels resembling common lymphatic malformation, and thoracic lesions can be related to a poor prognosis. Sirolimus, an inhibitor of the mammalian target of rapamycin, is effective aga...

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Detalles Bibliográficos
Autores principales: Fujii, Takayuki, Shimono, Ryuichi, Tanaka, Aya, Katami, Hiroto
Formato: Online Artículo Texto
Lenguaje:English
Publicado: Hindawi 2019
Materias:
Acceso en línea:https://www.ncbi.nlm.nih.gov/pmc/articles/PMC6515074/
https://www.ncbi.nlm.nih.gov/pubmed/31183237
http://dx.doi.org/10.1155/2019/3101357
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author Fujii, Takayuki
Shimono, Ryuichi
Tanaka, Aya
Katami, Hiroto
author_facet Fujii, Takayuki
Shimono, Ryuichi
Tanaka, Aya
Katami, Hiroto
author_sort Fujii, Takayuki
collection PubMed
description Generalized lymphatic anomaly (GLA) is characterized by diffuse or multicentric proliferation of dilated lymphatic vessels resembling common lymphatic malformation, and thoracic lesions can be related to a poor prognosis. Sirolimus, an inhibitor of the mammalian target of rapamycin, is effective against vascular anomalies with few severe adverse drug reactions. Here, we report the case of a patient with intractable hemothorax pleural effusion due to GLA who was treated with sirolimus and experienced disseminated intravascular coagulation. Although a standard treatment for GLA has not been established, pleural fluid might be reduced using the Kampo medicine Eppikajyutsuto.
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spelling pubmed-65150742019-06-10 A Case of Suspected Adverse Reactions to Sirolimus in the Treatment of Generalized Lymphatic Anomaly Fujii, Takayuki Shimono, Ryuichi Tanaka, Aya Katami, Hiroto Case Rep Pediatr Case Report Generalized lymphatic anomaly (GLA) is characterized by diffuse or multicentric proliferation of dilated lymphatic vessels resembling common lymphatic malformation, and thoracic lesions can be related to a poor prognosis. Sirolimus, an inhibitor of the mammalian target of rapamycin, is effective against vascular anomalies with few severe adverse drug reactions. Here, we report the case of a patient with intractable hemothorax pleural effusion due to GLA who was treated with sirolimus and experienced disseminated intravascular coagulation. Although a standard treatment for GLA has not been established, pleural fluid might be reduced using the Kampo medicine Eppikajyutsuto. Hindawi 2019-04-30 /pmc/articles/PMC6515074/ /pubmed/31183237 http://dx.doi.org/10.1155/2019/3101357 Text en Copyright © 2019 Takayuki Fujii et al. http://creativecommons.org/licenses/by/4.0/ This is an open access article distributed under the Creative Commons Attribution License, which permits unrestricted use, distribution, and reproduction in any medium, provided the original work is properly cited.
spellingShingle Case Report
Fujii, Takayuki
Shimono, Ryuichi
Tanaka, Aya
Katami, Hiroto
A Case of Suspected Adverse Reactions to Sirolimus in the Treatment of Generalized Lymphatic Anomaly
title A Case of Suspected Adverse Reactions to Sirolimus in the Treatment of Generalized Lymphatic Anomaly
title_full A Case of Suspected Adverse Reactions to Sirolimus in the Treatment of Generalized Lymphatic Anomaly
title_fullStr A Case of Suspected Adverse Reactions to Sirolimus in the Treatment of Generalized Lymphatic Anomaly
title_full_unstemmed A Case of Suspected Adverse Reactions to Sirolimus in the Treatment of Generalized Lymphatic Anomaly
title_short A Case of Suspected Adverse Reactions to Sirolimus in the Treatment of Generalized Lymphatic Anomaly
title_sort case of suspected adverse reactions to sirolimus in the treatment of generalized lymphatic anomaly
topic Case Report
url https://www.ncbi.nlm.nih.gov/pmc/articles/PMC6515074/
https://www.ncbi.nlm.nih.gov/pubmed/31183237
http://dx.doi.org/10.1155/2019/3101357
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