PUBLICATION
Expression analysis of genes including Zfhx4 in mice and zebrafish reveals a temporospatial conserved molecular basis underlying craniofacial development
- Authors
- Liu, S., Xu, L., Kashima, M., Narumi, R., Takahata, Y., Nakamura, E., Shibuya, H., Tamura, M., Shida, Y., Inubushi, T., Nukada, Y., Miyazawa, M., Hata, K., Nishimura, R., Yamashiro, T., Tasaki, J., Kurosaka, H.
- ID
- ZDB-PUB-240926-3
- Date
- 2024
- Source
- Developmental Dynamics : an official publication of the American Association of Anatomists : (Journal)
- Registered Authors
- Keywords
- cranial neural crest cells, disease models, loss‐of‐function analysis, palatogenesis, stage‐matched gene expression analysis, temporal expression patterns
- Datasets
- GEO:GSE254071
- MeSH Terms
-
- Gene Expression Regulation, Developmental*
- Animals
- Homeodomain Proteins*/genetics
- Homeodomain Proteins*/metabolism
- Mice
- Zebrafish*/embryology
- Zebrafish*/genetics
- Craniofacial Abnormalities/genetics
- Craniofacial Abnormalities/metabolism
- Zebrafish Proteins*/genetics
- Zebrafish Proteins*/metabolism
- PubMed
- 39320016 Full text @ Dev. Dyn.
Citation
Liu, S., Xu, L., Kashima, M., Narumi, R., Takahata, Y., Nakamura, E., Shibuya, H., Tamura, M., Shida, Y., Inubushi, T., Nukada, Y., Miyazawa, M., Hata, K., Nishimura, R., Yamashiro, T., Tasaki, J., Kurosaka, H. (2024) Expression analysis of genes including Zfhx4 in mice and zebrafish reveals a temporospatial conserved molecular basis underlying craniofacial development. Developmental Dynamics : an official publication of the American Association of Anatomists. :.
Abstract
Background Embryonic craniofacial development involves several cellular and molecular events that are evolutionarily conserved among vertebrates. Vertebrate models such as mice and zebrafish have been used to investigate the molecular and cellular etiologies underlying human craniofacial disorders, including orofacial clefts. However, the molecular mechanisms underlying embryonic development in these two species are unknown. Therefore, elucidating the shared mechanisms of craniofacial development between disease models is crucial to understanding the underlying mechanisms of phenotypes in individual species.
Results We selected mice and zebrafish as model organisms to compare various events during embryonic craniofacial development. We identified genes (Sox9, Zfhx3 and 4, Cjun, and Six1) exhibiting similar temporal expression patterns between these species through comprehensive and stage-matched gene expression analyses. Expression analysis revealed similar gene expression in hypothetically corresponding tissues, such as the mice palate and zebrafish ethmoid plate. Furthermore, loss-of-function analysis of Zfhx4/zfhx4, a causative gene of human craniofacial anomalies including orofacial cleft, in both species resulted in deformed skeletal elements such as the palatine and ethmoid plate in mice and zebrafish, respectively.
Conclusions These results demonstrate that these disease models share common molecular mechanisms, highlighting their usefulness in modeling craniofacial defects in humans.
Genes / Markers
Expression
Phenotype
Mutations / Transgenics
Human Disease / Model
Sequence Targeting Reagents
Fish
Orthology
Engineered Foreign Genes
Mapping