PUBLICATION

Unraveling the role of Ctla-4 in intestinal immune homeostasis through a novel Zebrafish model of inflammatory bowel disease

Authors
Qin, L., Hu, C., Zhao, Q., Wang, Y., Fan, D., Lin, A., Xiang, L., Chen, Y., Shao, J.
ID
ZDB-PUB-250521-2
Date
2025
Source
eLIFE   13: (Journal)
Registered Authors
Fan, Dongdong, Lin, Aifu, Shao, Jian-zhong
Keywords
Ctla-4 deficiency, immunology, inflammation, inflammatory bowel disease, zebrafish, zebrafish model
Datasets
GEO:GSE255304, GEO:GSE211396, GEO:GSE255303
MeSH Terms
  • Animals
  • Zebrafish Proteins*/genetics
  • Zebrafish Proteins*/metabolism
  • CTLA-4 Antigen*/genetics
  • CTLA-4 Antigen*/immunology
  • CTLA-4 Antigen*/metabolism
  • Disease Models, Animal
  • Intestines*/immunology
  • Zebrafish*/genetics
  • Zebrafish*/immunology
  • Inflammatory Bowel Diseases*/genetics
  • Inflammatory Bowel Diseases*/immunology
  • Inflammatory Bowel Diseases*/pathology
  • Gastrointestinal Microbiome
  • Homeostasis*
PubMed
40392591 Full text @ Elife
Abstract
Inflammatory bowel disease (IBD) is a chronic and relapsing immune-mediated disorder characterized by intestinal inflammation and epithelial injury. The underlying causes of IBD are not fully understood, but genetic factors have been implicated in genome-wide association studies, including CTLA-4, an essential negative regulator of T cell activation. However, establishing a direct link between CTLA-4 and IBD has been challenging due to the early lethality of CTLA-4 knockout mice. In this study, we identified zebrafish Ctla-4 homolog and investigated its role in maintaining intestinal immune homeostasis by generating a Ctla-4-deficient (ctla-4-/-) zebrafish line. These mutant zebrafish exhibited reduced weight, along with impaired epithelial barrier integrity and lymphocytic infiltration in their intestines. Transcriptomics analysis revealed upregulation of inflammation-related genes, disturbing immune system homeostasis. Moreover, single-cell RNA-sequencing analysis indicated increased Th2 cells and interleukin 13 expression, along with decreased innate lymphoid cells and upregulated proinflammatory cytokines. Additionally, Ctla-4-deficient zebrafish exhibited reduced diversity and an altered composition of the intestinal microbiota. All these phenotypes closely resemble those found in mammalian IBD. Lastly, supplementation with Ctla-4-Ig successfully alleviated intestinal inflammation in these mutants. Altogether, our findings demonstrate the pivotal role of Ctla-4 in maintaining intestinal homeostasis. Additionally, they offer substantial evidence linking CTLA-4 to IBD and establish a novel zebrafish model for investigating both the pathogenesis and potential treatments.
Genes / Markers
Figures
Show all Figures
Expression
Phenotype
Mutations / Transgenics
Human Disease / Model
Sequence Targeting Reagents
Fish
Antibodies
Orthology
Engineered Foreign Genes
Mapping