PUBLICATION
Incoherent collective cell chemotaxis underlies organ dysmorphia in a model of branchio-oto-renal syndrome
- Authors
- Borges, A., Pinto-Teixeira, F., Wibowo, I., Pogoda, H.M., Hammerschmidt, M., Kawakami, K., López-Schier, H., Miranda-Rodríguez, J.R.
- ID
- ZDB-PUB-241009-7
- Date
- 2024
- Source
- microPublication. Biology 2024: (Journal)
- Registered Authors
- Hammerschmidt, Matthias, Kawakami, Koichi, Pogoda, Hans-Martin
- Keywords
- none
- MeSH Terms
- none
- PubMed
- 39381636 Full text @ MicroPubl Biol
Citation
Borges, A., Pinto-Teixeira, F., Wibowo, I., Pogoda, H.M., Hammerschmidt, M., Kawakami, K., López-Schier, H., Miranda-Rodríguez, J.R. (2024) Incoherent collective cell chemotaxis underlies organ dysmorphia in a model of branchio-oto-renal syndrome. microPublication. Biology. 2024:.
Abstract
Mutations in eya1 cause branchio-oto-renal syndrome (BOR) in humans and the equivalent condition in animal models. BOR is characterized by multi-organ malformations. To better understand the role of Eya1 in organogenesis we used the zebrafish posterior lateral-line primordium. This multicellular tissue moves from head-to-tail at a constant velocity via the simultaneous action of two chemokine receptors, Cxcr4b and Ackr3b (formerly cxcr7b). We found that loss of eya1 strongly reduces the expression of ackr3b , disrupting the coherent motion of the primordium and leading to lateral-line truncations. These findings point to abnormal collective cell chemotaxis as the origin of organ dysmorphia in BOR.
Genes / Markers
Expression
Phenotype
Mutations / Transgenics
Human Disease / Model
Sequence Targeting Reagents
Fish
Orthology
Engineered Foreign Genes
Mapping