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
Abstract
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.
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.
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
Figures
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Expression
Phenotype
Mutations / Transgenics
Human Disease / Model
Sequence Targeting Reagents
Fish
Antibodies
Orthology
Engineered Foreign Genes
Mapping