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Tacrolimus in Gastrointestinal Bleeding in a Young Boy With Hereditary Hemorrhagic Telangiectasia

Hereditary hemorrhagic telangiectasia (HHT) is an autosomal dominant disease in which gastrointestinal bleeding is a rare presenting symptom in children. Gastrointestinal bleeding in children is treated locally by endoscopy. When a focus of bleeding cannot be reached by endoscopy, management of thes...

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Autores principales: Pruijsen, Jessica M., Kroon, Steven, Mager, Johannes J., Bungener, Laura B., van der Doef, Hubert P. J.
Formato: Online Artículo Texto
Lenguaje:English
Publicado: Lippincott Williams & Wilkins, Inc. 2021
Materias:
Acceso en línea:https://www.ncbi.nlm.nih.gov/pmc/articles/PMC10191567/
https://www.ncbi.nlm.nih.gov/pubmed/37206467
http://dx.doi.org/10.1097/PG9.0000000000000133
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author Pruijsen, Jessica M.
Kroon, Steven
Mager, Johannes J.
Bungener, Laura B.
van der Doef, Hubert P. J.
author_facet Pruijsen, Jessica M.
Kroon, Steven
Mager, Johannes J.
Bungener, Laura B.
van der Doef, Hubert P. J.
author_sort Pruijsen, Jessica M.
collection PubMed
description Hereditary hemorrhagic telangiectasia (HHT) is an autosomal dominant disease in which gastrointestinal bleeding is a rare presenting symptom in children. Gastrointestinal bleeding in children is treated locally by endoscopy. When a focus of bleeding cannot be reached by endoscopy, management of these patients can be challenging. Previous reports showed a favorable outcome of treatment with tacrolimus in an adult HHT patient with liver vascular malformations and epistaxis and in a HHT patient with pulmonary hypertension. We report the first pediatric HHT patient who benefited from tacrolimus treatment. Our case demonstrated a remarkable decline in blood transfusions and better quality of life during the period of tacrolimus treatment.
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spelling pubmed-101915672023-05-18 Tacrolimus in Gastrointestinal Bleeding in a Young Boy With Hereditary Hemorrhagic Telangiectasia Pruijsen, Jessica M. Kroon, Steven Mager, Johannes J. Bungener, Laura B. van der Doef, Hubert P. J. JPGN Rep Case Report Hereditary hemorrhagic telangiectasia (HHT) is an autosomal dominant disease in which gastrointestinal bleeding is a rare presenting symptom in children. Gastrointestinal bleeding in children is treated locally by endoscopy. When a focus of bleeding cannot be reached by endoscopy, management of these patients can be challenging. Previous reports showed a favorable outcome of treatment with tacrolimus in an adult HHT patient with liver vascular malformations and epistaxis and in a HHT patient with pulmonary hypertension. We report the first pediatric HHT patient who benefited from tacrolimus treatment. Our case demonstrated a remarkable decline in blood transfusions and better quality of life during the period of tacrolimus treatment. Lippincott Williams & Wilkins, Inc. 2021-10-25 /pmc/articles/PMC10191567/ /pubmed/37206467 http://dx.doi.org/10.1097/PG9.0000000000000133 Text en Copyright © 2021 The Author(s). Published by Wolters Kluwer on behalf of European Society for Pediatric Gastroenterology, Hepatology, and Nutrition and North American Society for Pediatric Gastroenterology, Hepatology, and Nutrition. https://creativecommons.org/licenses/by-nc-nd/4.0/This is an open-access article distributed under the terms of the Creative Commons Attribution-Non Commercial-No Derivatives License 4.0 (CCBY-NC-ND) (https://creativecommons.org/licenses/by-nc-nd/4.0/) , where it is permissible to download and share the work provided it is properly cited. The work cannot be changed in any way or used commercially without permission from the journal.
spellingShingle Case Report
Pruijsen, Jessica M.
Kroon, Steven
Mager, Johannes J.
Bungener, Laura B.
van der Doef, Hubert P. J.
Tacrolimus in Gastrointestinal Bleeding in a Young Boy With Hereditary Hemorrhagic Telangiectasia
title Tacrolimus in Gastrointestinal Bleeding in a Young Boy With Hereditary Hemorrhagic Telangiectasia
title_full Tacrolimus in Gastrointestinal Bleeding in a Young Boy With Hereditary Hemorrhagic Telangiectasia
title_fullStr Tacrolimus in Gastrointestinal Bleeding in a Young Boy With Hereditary Hemorrhagic Telangiectasia
title_full_unstemmed Tacrolimus in Gastrointestinal Bleeding in a Young Boy With Hereditary Hemorrhagic Telangiectasia
title_short Tacrolimus in Gastrointestinal Bleeding in a Young Boy With Hereditary Hemorrhagic Telangiectasia
title_sort tacrolimus in gastrointestinal bleeding in a young boy with hereditary hemorrhagic telangiectasia
topic Case Report
url https://www.ncbi.nlm.nih.gov/pmc/articles/PMC10191567/
https://www.ncbi.nlm.nih.gov/pubmed/37206467
http://dx.doi.org/10.1097/PG9.0000000000000133
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