Observational Study
Copyright ©The Author(s) 2016. Published by Baishideng Publishing Group Inc. All rights reserved.
World J Gastroenterol. Dec 28, 2016; 22(48): 10653-10662
Published online Dec 28, 2016. doi: 10.3748/wjg.v22.i48.10653
Variable outcome in infantile-onset inflammatory bowel disease in an Asian cohort
Way Seah Lee, Ruey Terng Ng, Koon-Wing Chan, Yu-Lung Lau
Way Seah Lee, Ruey Terng Ng, Department of Paediatrics, University Malaya Medical Center, Kuala Lumpur 59100, Malaysia
Way Seah Lee, University Malaya Paediatric and Child Health Research Group, University Malaya, Kuala Lumpur 59100, Malaysia
Koon-Wing Chan, Yu-Lung Lau, Department of Paediatrics and Adolescent Medicine, Queen Mary Hospital, LKS Faculty of Medicine, The University of Hong Kong, Hong Kong, China
Author contributions: Lee WS conceived the concept and wrote the first draft; both Lee WS and Ng RT collected the clinical data and performed data analysis; Chan KW and Lau YL performed the mutational analysis; Lau YL provided critical analysis of the manuscript.
Supported by a research grant from Ministry of Higher Education, Malaysia (UM.C/625/HIR/MOHE/CHAN/13/1); Hong Kong Society for the Relief of Disabled Children (to Chan KW and Lau YL).
Institutional review board statement: The present study was reviewed and approved by the Medical Ethics Committee of University Malaya Medical Centre, Kuala Lumpur, Malaysia.
Informed consent statement: The legal guardians of all patients described in the present study gave informed written consent for mutational analysis performed for the present study.
Conflict-of-interest statement: The authors have declared that no competing interests exist.
Open-Access: 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/
Correspondence to: Dr. Way Seah Lee, Professor, Department of Paediatrics, University Malaya Medical Center, Lembah Pantai, Kuala Lumpur 59100, Malaysia. leews@um.edu.my
Telephone: +66-603-79492065 Fax: +66-603-79494704
Received: March 5, 2016
Peer-review started: March 8, 2016
First decision: April 14, 2016
Revised: August 31, 2016
Accepted: October 19, 2016
Article in press: October 19, 2016
Published online: December 28, 2016
Processing time: 295 Days and 19.5 Hours
Abstract
AIM

Infantile-onset inflammatory bowel disease (IO-IBD) with the onset of disease before 12 mo of age, is a different disease entity from childhood IBD. We aimed to describe the clinical features, outcome and role of mutation in interleukin-10 (IL-10) and interleukin-10 receptors (IL-10R) in Asian children with IO-IBD.

METHODS

All cases of IO-IBD, defined as onset of disease before 12 mo of age, seen at University Malaya Medical Center, Malaysia were reviewed. We performed mutational analysis for IL10 and IL10R genes in patients with presenting clinical features of Crohn’s disease (CD).

RESULTS

Six [13%; CD = 3, ulcerative colitis (UC) = 2, IBD-unclassified (IBD-U) = 1] of the 48 children (CD = 25; UC = 23) with IBD have IO-IBD. At final review [median (range) duration of follow-up: 6.5 (3.0-20) years], three patients were in remission without immunosuppression [one each for post-colostomy (IBD-U), after standard immunosuppression (CD), and after total colectomy (UC)]. Three patients were on immunosuppression: one (UC) was in remission while two (both CD) had persistent disease. As compared with later-onset disease, IO-IBD were more likely to present with bloody diarrhea (100% vs 55%, P = 0.039) but were similar in terms of an associated autoimmune liver disease (0% vs 19%, P = 0.31), requiring biologics therapy (50% vs 36%, P = 0.40), surgery (50% vs 29%, P = 0.27), or achieving remission (50% vs 64%, P = 0.40). No mutations in either IL10 or IL10R in the three patients with CD and the only patient with IBD-U were identified.

CONCLUSION

The clinical features of IO-IBD in this Asian cohort of children who were negative for IL-10 or IL-10R mutations were variable. As compared to childhood IBD with onset of disease after 12 mo of age, IO-IBD achieved remission at a similar rate.

Keywords: Infantile-onset inflammatory bowel disease; Pediatric

Core tip: We described the clinical features, outcome and role of mutation in IL-10 and IL-10R in Asian children with infantile-onset inflammatory bowel disease (IO-IBD). We reviewed all cases of IO-IBD, defined as onset of disease before 12 mo of age, seen at a single center in Malaysia. We conclude that the clinical features of IO-IBD in this Asian cohort of children were variable. IO-IBD achieved remission at a similar rate, were more likely to discontinue immunosuppression therapy at final review and not more likely to require biologics therapy or surgery.