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Juvenile polyposis syndrome: An unusual case report of anemia and gastrointestinal bleeding in young infant

BACKGROUND: Juvenile polyposis syndrome, a rare disorder in children, is characterized with multiple hamartomatous polyps in alimentary tract. A variety of manifestations include bleeding, intussusception, or polyp prolapse. In this study, we present an 8-month-old male infant of juvenile polyposis...

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Autores principales: Hsiao, Yi-Han, Wei, Chin-Hung, Chang, Szu-Wen, Chang, Lung, Fu, Yu-Wei, Lee, Hung-Chang, Liu, Hsuan-Liang, Yeung, Chun-Yan
Formato: Online Artículo Texto
Lenguaje:English
Publicado: Wolters Kluwer Health 2016
Materias:
Acceso en línea:https://www.ncbi.nlm.nih.gov/pmc/articles/PMC5402548/
https://www.ncbi.nlm.nih.gov/pubmed/27631205
http://dx.doi.org/10.1097/MD.0000000000004550
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author Hsiao, Yi-Han
Wei, Chin-Hung
Chang, Szu-Wen
Chang, Lung
Fu, Yu-Wei
Lee, Hung-Chang
Liu, Hsuan-Liang
Yeung, Chun-Yan
author_facet Hsiao, Yi-Han
Wei, Chin-Hung
Chang, Szu-Wen
Chang, Lung
Fu, Yu-Wei
Lee, Hung-Chang
Liu, Hsuan-Liang
Yeung, Chun-Yan
author_sort Hsiao, Yi-Han
collection PubMed
description BACKGROUND: Juvenile polyposis syndrome, a rare disorder in children, is characterized with multiple hamartomatous polyps in alimentary tract. A variety of manifestations include bleeding, intussusception, or polyp prolapse. In this study, we present an 8-month-old male infant of juvenile polyposis syndrome initially presenting with chronic anemia. To the best of our knowledge, this is the youngest case reported in the literature. METHODS: We report a rare case of an 8-month-old male infant who presented with chronic anemia and gastrointestinal bleeding initially. Panendoscopy and abdominal computed tomography showed multiple polyposis throughout the entire alimentary tract leading to intussusception. Technetium-99m-labeled red blood cell (RBC) bleeding scan revealed the possibility of gastrointestinal tract bleeding in the jejunum. Histopathological examination on biopsy samples showed Peutz-Jeghers syndrome was excluded, whereas the diagnosis of juvenile polyposis syndrome was established. RESULTS: Enteroscopic polypectomy is the mainstay of the treatment. However, polyps recurred and occupied the majority of the gastrointestinal tract in 6 months. Supportive management was given. The patient expired for severe sepsis at the age of 18 months. CONCLUSION: Juvenile polyposis syndrome is an inherited disease, so it is not possible to prevent it. Concerning of its poor outcome and high mortality rate, it is important that we should increase awareness and education of the parents at its earliest stages.
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spelling pubmed-54025482017-04-27 Juvenile polyposis syndrome: An unusual case report of anemia and gastrointestinal bleeding in young infant Hsiao, Yi-Han Wei, Chin-Hung Chang, Szu-Wen Chang, Lung Fu, Yu-Wei Lee, Hung-Chang Liu, Hsuan-Liang Yeung, Chun-Yan Medicine (Baltimore) 6200 BACKGROUND: Juvenile polyposis syndrome, a rare disorder in children, is characterized with multiple hamartomatous polyps in alimentary tract. A variety of manifestations include bleeding, intussusception, or polyp prolapse. In this study, we present an 8-month-old male infant of juvenile polyposis syndrome initially presenting with chronic anemia. To the best of our knowledge, this is the youngest case reported in the literature. METHODS: We report a rare case of an 8-month-old male infant who presented with chronic anemia and gastrointestinal bleeding initially. Panendoscopy and abdominal computed tomography showed multiple polyposis throughout the entire alimentary tract leading to intussusception. Technetium-99m-labeled red blood cell (RBC) bleeding scan revealed the possibility of gastrointestinal tract bleeding in the jejunum. Histopathological examination on biopsy samples showed Peutz-Jeghers syndrome was excluded, whereas the diagnosis of juvenile polyposis syndrome was established. RESULTS: Enteroscopic polypectomy is the mainstay of the treatment. However, polyps recurred and occupied the majority of the gastrointestinal tract in 6 months. Supportive management was given. The patient expired for severe sepsis at the age of 18 months. CONCLUSION: Juvenile polyposis syndrome is an inherited disease, so it is not possible to prevent it. Concerning of its poor outcome and high mortality rate, it is important that we should increase awareness and education of the parents at its earliest stages. Wolters Kluwer Health 2016-09-16 /pmc/articles/PMC5402548/ /pubmed/27631205 http://dx.doi.org/10.1097/MD.0000000000004550 Text en Copyright © 2016 the Author(s). Published by Wolters Kluwer Health, Inc. All rights reserved. http://creativecommons.org/licenses/by-nc/4.0 This is an open-access article distributed under the terms of the Creative Commons Attribution-Non Commercial License 4.0 (CCBY-NC), where it is permissible to download, share, remix, transform, and buildup the work provided it is properly cited. The work cannot be used commercially. http://creativecommons.org/licenses/by-nc/4.0
spellingShingle 6200
Hsiao, Yi-Han
Wei, Chin-Hung
Chang, Szu-Wen
Chang, Lung
Fu, Yu-Wei
Lee, Hung-Chang
Liu, Hsuan-Liang
Yeung, Chun-Yan
Juvenile polyposis syndrome: An unusual case report of anemia and gastrointestinal bleeding in young infant
title Juvenile polyposis syndrome: An unusual case report of anemia and gastrointestinal bleeding in young infant
title_full Juvenile polyposis syndrome: An unusual case report of anemia and gastrointestinal bleeding in young infant
title_fullStr Juvenile polyposis syndrome: An unusual case report of anemia and gastrointestinal bleeding in young infant
title_full_unstemmed Juvenile polyposis syndrome: An unusual case report of anemia and gastrointestinal bleeding in young infant
title_short Juvenile polyposis syndrome: An unusual case report of anemia and gastrointestinal bleeding in young infant
title_sort juvenile polyposis syndrome: an unusual case report of anemia and gastrointestinal bleeding in young infant
topic 6200
url https://www.ncbi.nlm.nih.gov/pmc/articles/PMC5402548/
https://www.ncbi.nlm.nih.gov/pubmed/27631205
http://dx.doi.org/10.1097/MD.0000000000004550
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