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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...
Autores principales: | , , , , , , , |
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Formato: | Online Artículo Texto |
Lenguaje: | English |
Publicado: |
Wolters Kluwer Health
2016
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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. |
format | Online Article Text |
id | pubmed-5402548 |
institution | National Center for Biotechnology Information |
language | English |
publishDate | 2016 |
publisher | Wolters Kluwer Health |
record_format | MEDLINE/PubMed |
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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