Gong YZ, Zhong XM, Zou JZ. Infliximab treatment of glycogenosis Ib with Crohn's-like enterocolitis: A case report. World J Clin Cases 2021; 9(19): 5280-5286 [PMID: 34307579 DOI: 10.12998/wjcc.v9.i19.5280]
Corresponding Author of This Article
Xue-Mei Zhong, MD, Associate Professor, Chief Doctor, Gastro-enterology Department, Capital Institute of Pediatrics, No. 2 Yabao Road, Chaoyang District, Beijing 100020, China. zhongxuemei@shouer.com.cn
Research Domain of This Article
Gastroenterology & Hepatology
Article-Type of This Article
Case Report
Open-Access Policy of This Article
This article is an open-access article which was selected by an in-house editor and fully peer-reviewed by external reviewers. It is distributed in accordance with the Creative Commons Attribution Non Commercial (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/
World J Clin Cases. Jul 6, 2021; 9(19): 5280-5286 Published online Jul 6, 2021. doi: 10.12998/wjcc.v9.i19.5280
Infliximab treatment of glycogenosis Ib with Crohn's-like enterocolitis: A case report
You-Zhe Gong, Xue-Mei Zhong, Ji-Zhen Zou
You-Zhe Gong, Xue-Mei Zhong, Gastroenterology Department, Capital Institute of Pediatrics, Beijing 100020, China
Ji-Zhen Zou, Pathology Department, Capital Institute of Pediatrics, Beijing 100020, China
Author contributions: Gong YZ carried out the study, participated in collecting data, and drafted the manuscript; Zhong XM conceived the study and revised the paper; Zou JZ provided pathological data and revised the paper; All authors read and approved the final manuscript.
Supported bythe Digestive Medical Coordinated Development Center of Beijing Hospitals Authority, No.XXZ0505.
Informed consent statement: Informed written consent was obtained from the patient for publication of this case report and accompanying images.
Conflict-of-interest statement: The authors declare that they have no competing interests.
CARE Checklist (2016) statement: The authors have read the CARE Checklist (2016), and the manuscript was prepared and revised according to the CARE Checklist.
Open-Access: 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/
Corresponding author: Xue-Mei Zhong, MD, Associate Professor, Chief Doctor, Gastro-enterology Department, Capital Institute of Pediatrics, No. 2 Yabao Road, Chaoyang District, Beijing 100020, China. zhongxuemei@shouer.com.cn
Received: February 9, 2021 Peer-review started: February 9, 2021 First decision: February 28, 2021 Revised: March 11, 2021 Accepted: April 25, 2021 Article in press: April 25, 2021 Published online: July 6, 2021 Processing time: 134 Days and 21.3 Hours
Abstract
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.
Core Tip: Conventional treatment cannot alleviate symptoms of intestinal inflammation in glycogen storage disease type Ib (GSD-Ib). Although biological agents are effective for treating Crohn’s disease (CD), their application in the treatment of GSD-Ib with CD has been rarely reported. Infliximab was selected for this patient, and the intestinal symptoms were successfully alleviated. For cases with poor outcome using the granulocyte colony-stimulating factor treatment, infliximab can be used for therapy.
