PUBLICATION
VPS13B is localized at the interface between Golgi cisternae and is a functional partner of FAM177A1
- Authors
- Ugur, B., Schueder, F., Shin, J., Hanna, M.G., Wu, Y., Leonzino, M., Su, M., McAdow, A.R., Wilson, C., Postlethwait, J., Solnica-Krezel, L., Bewersdorf, J., De Camilli, P.
- ID
- ZDB-PUB-240928-7
- Date
- 2024
- Source
- The Journal of cell biology 223(12): (Journal)
- Registered Authors
- Postlethwait, John H., Shin, Jimann, Solnica-Krezel, Lilianna, Wilson, Catherine
- Keywords
- none
- MeSH Terms
-
- HeLa Cells
- Zebrafish Proteins/genetics
- Zebrafish Proteins/metabolism
- Golgi Apparatus*/metabolism
- Animals
- Protein Binding
- Zebrafish*/genetics
- Brefeldin A/pharmacology
- Humans
- Protein Transport
- Vesicular Transport Proteins*/genetics
- Vesicular Transport Proteins*/metabolism
- PubMed
- 39331042 Full text @ J. Cell Biol.
Citation
Ugur, B., Schueder, F., Shin, J., Hanna, M.G., Wu, Y., Leonzino, M., Su, M., McAdow, A.R., Wilson, C., Postlethwait, J., Solnica-Krezel, L., Bewersdorf, J., De Camilli, P. (2024) VPS13B is localized at the interface between Golgi cisternae and is a functional partner of FAM177A1. The Journal of cell biology. 223(12):.
Abstract
Mutations in VPS13B, a member of a protein family implicated in bulk lipid transport between adjacent membranes, cause Cohen syndrome. VPS13B is known to be concentrated in the Golgi complex, but its precise location within this organelle and thus the site(s) where it achieves lipid transport remains unclear. Here, we show that VPS13B is localized at the interface between proximal and distal Golgi subcompartments and that Golgi complex reformation after Brefeldin A (BFA)-induced disruption is delayed in VPS13B KO cells. This delay is phenocopied by the loss of FAM177A1, a Golgi complex protein of unknown function reported to be a VPS13B interactor and whose mutations also result in a developmental disorder. In zebrafish, the vps13b ortholog, not previously annotated in this organism, genetically interacts with fam177a1. Collectively, these findings raise the possibility that bulk lipid transport by VPS13B may play a role in the dynamics of Golgi membranes and that VPS13B may be assisted in this function by FAM177A1.
Genes / Markers
Expression
Phenotype
Mutations / Transgenics
Human Disease / Model
Sequence Targeting Reagents
Fish
Orthology
Engineered Foreign Genes
Mapping