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Infliximab treatment of glycogenosis Ib with Crohn's-like enterocolitis: A case report

BACKGROUND: Glycogen storage disease type Ib (GSD-Ib) is a glycogen metabolism disorder that leads to the manifestations of inflammatory bowel disease (IBD), especially Crohn’s disease (CD)-like colitis. Although biological agents are effective for treating CD, their application in the treatment of...

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Autores principales: Gong, You-Zhe, Zhong, Xue-Mei, Zou, Ji-Zhen
Formato: Online Artículo Texto
Lenguaje:English
Publicado: Baishideng Publishing Group Inc 2021
Materias:
Acceso en línea:https://www.ncbi.nlm.nih.gov/pmc/articles/PMC8283598/
https://www.ncbi.nlm.nih.gov/pubmed/34307579
http://dx.doi.org/10.12998/wjcc.v9.i19.5280
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author Gong, You-Zhe
Zhong, Xue-Mei
Zou, Ji-Zhen
author_facet Gong, You-Zhe
Zhong, Xue-Mei
Zou, Ji-Zhen
author_sort Gong, You-Zhe
collection PubMed
description BACKGROUND: Glycogen storage disease type Ib (GSD-Ib) is a glycogen metabolism disorder that leads to the manifestations of inflammatory bowel disease (IBD), especially Crohn’s disease (CD)-like colitis. Although biological agents are effective for treating CD, their application in the treatment of GSD-Ib with CD-like colitis has been rarely reported. CASE SUMMARY: A 13-year-old Han male was diagnosed with GSD-Ib with CD. The patient was treated with granulocyte colony-stimulating factor. When he had symptoms of CD-like colitis, he was continuously pumped with enteral nutrition and administered oral mesalazine for 2 wk; however, the symptoms did not improve significantly. Hence, infliximab (IFX) was administered. Hitherto, the patient has been followed up for 1 year, and no clinical manifestations have been observed. After 6 mo of treatment (fifth IFX treatment), the disease activity index and all inflammatory indexes decreased, and a review of the colonoscopy data showed that the ulcers appeared smooth. CONCLUSION: In this study, the patient was successfully treated with IFX. In cases of GSD-Ib, IBD should be highly considered.
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spelling pubmed-82835982021-07-23 Infliximab treatment of glycogenosis Ib with Crohn's-like enterocolitis: A case report Gong, You-Zhe Zhong, Xue-Mei Zou, Ji-Zhen World J Clin Cases Case Report BACKGROUND: Glycogen storage disease type Ib (GSD-Ib) is a glycogen metabolism disorder that leads to the manifestations of inflammatory bowel disease (IBD), especially Crohn’s disease (CD)-like colitis. Although biological agents are effective for treating CD, their application in the treatment of GSD-Ib with CD-like colitis has been rarely reported. CASE SUMMARY: A 13-year-old Han male was diagnosed with GSD-Ib with CD. The patient was treated with granulocyte colony-stimulating factor. When he had symptoms of CD-like colitis, he was continuously pumped with enteral nutrition and administered oral mesalazine for 2 wk; however, the symptoms did not improve significantly. Hence, infliximab (IFX) was administered. Hitherto, the patient has been followed up for 1 year, and no clinical manifestations have been observed. After 6 mo of treatment (fifth IFX treatment), the disease activity index and all inflammatory indexes decreased, and a review of the colonoscopy data showed that the ulcers appeared smooth. CONCLUSION: In this study, the patient was successfully treated with IFX. In cases of GSD-Ib, IBD should be highly considered. Baishideng Publishing Group Inc 2021-07-06 2021-07-06 /pmc/articles/PMC8283598/ /pubmed/34307579 http://dx.doi.org/10.12998/wjcc.v9.i19.5280 Text en ©The Author(s) 2021. Published by Baishideng Publishing Group Inc. All rights reserved. https://creativecommons.org/licenses/by-nc/4.0/This article is an open-access article that was selected by an in-house editor and fully peer-reviewed by external reviewers. It is distributed in accordance with the Creative Commons Attribution NonCommercial (CC BY-NC 4.0) license, which permits others to distribute, remix, adapt, build upon this work non-commercially, and license their derivative works on different terms, provided the original work is properly cited and the use is non-commercial. See: http://creativecommons.org/Licenses/by-nc/4.0/
spellingShingle Case Report
Gong, You-Zhe
Zhong, Xue-Mei
Zou, Ji-Zhen
Infliximab treatment of glycogenosis Ib with Crohn's-like enterocolitis: A case report
title Infliximab treatment of glycogenosis Ib with Crohn's-like enterocolitis: A case report
title_full Infliximab treatment of glycogenosis Ib with Crohn's-like enterocolitis: A case report
title_fullStr Infliximab treatment of glycogenosis Ib with Crohn's-like enterocolitis: A case report
title_full_unstemmed Infliximab treatment of glycogenosis Ib with Crohn's-like enterocolitis: A case report
title_short Infliximab treatment of glycogenosis Ib with Crohn's-like enterocolitis: A case report
title_sort infliximab treatment of glycogenosis ib with crohn's-like enterocolitis: a case report
topic Case Report
url https://www.ncbi.nlm.nih.gov/pmc/articles/PMC8283598/
https://www.ncbi.nlm.nih.gov/pubmed/34307579
http://dx.doi.org/10.12998/wjcc.v9.i19.5280
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