PUBLICATION
Genetic and protein interaction studies between the ciliary dyslexia candidate genes DYX1C1 and DCDC2
- Authors
- Bieder, A., Chandrasekar, G., Wason, A., Erkelenz, S., Gopalakrishnan, J., Kere, J., Tapia-Páez, I.
- ID
- ZDB-PUB-230528-38
- Date
- 2023
- Source
- BMC molecular and cell biology 24: 2020 (Journal)
- Registered Authors
- Keywords
- Centrosome, Cilia, Dyslexia, Genetic interaction, Zebrafish
- MeSH Terms
-
- Animals
- Cell Movement/genetics
- Cilia*/pathology
- Gene Expression Regulation
- Molecular Chaperones*/genetics
- Phenotype
- Zebrafish*/genetics
- Zebrafish Proteins*/genetics
- PubMed
- 37237337 Full text @ BMC Mol Cell Biol
Citation
Bieder, A., Chandrasekar, G., Wason, A., Erkelenz, S., Gopalakrishnan, J., Kere, J., Tapia-Páez, I. (2023) Genetic and protein interaction studies between the ciliary dyslexia candidate genes DYX1C1 and DCDC2. BMC molecular and cell biology. 24:2020.
Abstract
Background DYX1C1 (DNAAF4) and DCDC2 are two of the most replicated dyslexia candidate genes in genetic studies. They both have demonstrated roles in neuronal migration, in cilia growth and function and they both are cytoskeletal interactors. In addition, they both have been characterized as ciliopathy genes. However, their exact molecular functions are still incompletely described. Based on these known roles, we asked whether DYX1C1 and DCDC2 interact on the genetic and the protein level.
Results Here, we report the physical protein-protein interaction of DYX1C1 and DCDC2 as well as their respective interactions with the centrosomal protein CPAP (CENPJ) on exogenous and endogenous levels in different cell models including brain organoids. In addition, we show a synergistic genetic interaction between dyx1c1 and dcdc2b in zebrafish exacerbating the ciliary phenotype. Finally, we show a mutual effect on transcriptional regulation among DYX1C1 and DCDC2 in a cellular model.
Conclusions In summary, we describe the physical and functional interaction between the two genes DYX1C1 and DCDC2. These results contribute to the growing understanding of the molecular roles of DYX1C1 and DCDC2 and set the stage for future functional studies.
Genes / Markers
Expression
Phenotype
Mutations / Transgenics
Human Disease / Model
Sequence Targeting Reagents
Fish
Orthology
Engineered Foreign Genes
Mapping