PUBLICATION
Myosin 1b regulates intestinal epithelial morphogenesis via interaction with UNC45A
- Authors
- Revenu, C., Lebreton, C., Cannata Serio, M., Rosello, M., Duclaux-Loras, R., Duroure, K., Nicolle, O., Eggeler, F., Prospéri, M.T., Stoufflet, J., Vougny, J., Lépine, P., Michaux, G., Cerf-Bensussan, N., Coudrier, E., Perez, F., Parlato, M., Del Bene, F.
- ID
- ZDB-PUB-241206-2
- Date
- 2024
- Source
- Cell Reports 43: 114941114941 (Journal)
- Registered Authors
- Del Bene, Filippo, Duroure, Karine, Eggeler, Fanny, Revenu, Celine, Rosello, Marion
- Keywords
- CP: Cell biology, CP: Developmental biology, Intestinal epithelium, MYO1B, MYO5B, UNC45A, lumenogenesis, zebrafish
- MeSH Terms
-
- Humans
- Animals
- Intestinal Mucosa*/metabolism
- Intestinal Mucosa*/pathology
- Zebrafish*/metabolism
- Myosin Type I*/genetics
- Myosin Type I*/metabolism
- Morphogenesis*
- Zebrafish Proteins/genetics
- Zebrafish Proteins/metabolism
- Protein Binding
- Caco-2 Cells
- PubMed
- 39636728 Full text @ Cell Rep.
Citation
Revenu, C., Lebreton, C., Cannata Serio, M., Rosello, M., Duclaux-Loras, R., Duroure, K., Nicolle, O., Eggeler, F., Prospéri, M.T., Stoufflet, J., Vougny, J., Lépine, P., Michaux, G., Cerf-Bensussan, N., Coudrier, E., Perez, F., Parlato, M., Del Bene, F. (2024) Myosin 1b regulates intestinal epithelial morphogenesis via interaction with UNC45A. Cell Reports. 43:114941114941.
Abstract
Vesicle trafficking and the establishment of apicobasal polarity are essential processes in epithelial morphogenesis. UNC45A deficiency has been reported in a multi-organ syndrome presenting with severe diarrhea associated with enterocyte polarity defects. Myosin 1b, an actin motor able to bind membranes, regulates membrane shaping and vesicle trafficking. Here, we show that MYO1B is part of the UNC45A interactome. In the absence of UNC45A, myosin 1b is degraded and forms aggregates when proteasome activity is inhibited. In 3D Caco-2 cells, lumen formation is impaired in the absence of myosin 1b, associated with spindle orientation defects, Golgi apparatus fragmentation, and trafficking impairment. In zebrafish larvae, loss of myo1b results in intestinal bulb epithelium folding defects associated with terminal web disorganization and vesicle accumulation, reminiscent of villous atrophy. In conclusion, we show that myosin 1b plays an unexpected role in the development of the intestinal epithelium downstream of UNC45A, establishing its contribution in the gut defects reported in UNC45A patients.
Genes / Markers
Expression
Phenotype
Mutations / Transgenics
Human Disease / Model
Sequence Targeting Reagents
Fish
Orthology
Engineered Foreign Genes
Mapping