PUBLICATION
C-mannosyltransferase DPY19L1L-mediated Reissner Fiber formation is critical for zebrafish (Danio rerio) body axis straightening
- Authors
- Tian, G., Huang, L., Xu, Z., Lu, C., Yuan, W., Wu, Y., Liao, Z., Gao, J., Luo, Q., Cheng, B., Liao, X., Lu, H.
- ID
- ZDB-PUB-250510-12
- Date
- 2025
- Source
- Science advances 11: eadv2032eadv2032 (Journal)
- Registered Authors
- Lu, Huiqiang
- Keywords
- none
- MeSH Terms
-
- Animals
- Gene Expression Regulation, Developmental
- Mutation
- Body Patterning*/genetics
- Zebrafish*/embryology
- Zebrafish*/genetics
- Zebrafish*/metabolism
- Zebrafish Proteins*/genetics
- Zebrafish Proteins*/metabolism
- Embryo, Nonmammalian/metabolism
- Mannosyltransferases*/genetics
- Mannosyltransferases*/metabolism
- PubMed
- 40344050 Full text @ Sci Adv
Citation
Tian, G., Huang, L., Xu, Z., Lu, C., Yuan, W., Wu, Y., Liao, Z., Gao, J., Luo, Q., Cheng, B., Liao, X., Lu, H. (2025) C-mannosyltransferase DPY19L1L-mediated Reissner Fiber formation is critical for zebrafish (Danio rerio) body axis straightening. Science advances. 11:eadv2032eadv2032.
Abstract
The successful secretion and assembly of subcommissural organ (SCO)-spondin are crucial for Reissner Fiber (RF) formation and body axis straightening in zebrafish. However, the mechanisms underlying RF formation remain largely unknown. Here, we report that the C-mannosyltransferase dpy19l1l (dumpy-19 like 1 like) is expressed in the spinal cord during zebrafish embryonic development. Mutation in dpy19l1l resulted in idiopathic scoliosis (IS)-like body axis curvature in the absence of muscle or cilia defects. URP2 expression was down-regulated in dpy19l1l-/- mutants. Notably, RF formation was impaired in dpy19l1l-/- mutants, and a similar phenotype was induced in wild-type embryos by injecting messenger RNA encoding a C-mannosylation catalytic site-mutated dpy19l1l variant (E106A mdpy19l1l). Furthermore, E106A mDPY19L1L failed to glycosylate Flag-tagged SCO-spondin TSRs (thrombospondin type 1 repeats). Our findings suggest that DPY19L1L-mediated C-mannosylation of SCO-spondin TSRs promotes RF formation and URP2 induction, representing a critical supplementary mechanism for body axis straightening in zebrafish.
Genes / Markers
Expression
Phenotype
Mutations / Transgenics
Human Disease / Model
Sequence Targeting Reagents
Fish
Orthology
Engineered Foreign Genes
Mapping