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titleExpression analysis of genes including Zfhx4 in mice and zebrafish reveals a temporospatial conserved molecular basis underlying craniofacial development
DOIhttps://doi.org/10.1002/dvdy.740
Full Text URIhttps://ir.library.osaka-u.ac.jp/repo/ouka/all/103547/DevDyn_254_3_257.pdf
Author(eng)Liu, Shujie; Xu, Lin; Kashima, Makoto; Narumi, Rika; Takahata, Yoshifumi; Nakamura, Eriko; Shibuya, Hirotoshi; Tamura, Masaru; Shida, Yuki; Inubushi, Toshihiro; Nukada, Yuko; Miyazawa, Masaaki; Hata, Kenji; Nishimura, Riko; Yamashiro, Takashi; Tasaki, Junichi; Kurosaka, Hiroshi
Issue Date2024-09-25
PublisherJohn Wiley and Sons Inc
Publication titleDevelopmental Dynamics
Volume254
Issue3
Start page257
End page271
Languageeng
DescriptionLiu S., Xu L., Kashima M., et al. Expression analysis of genes including Zfhx4 in mice and zebrafish reveals a temporospatial conserved molecular basis underlying craniofacial development. Developmental Dynamics 254, 257 (2025); https://doi.org/10.1002/dvdy.740.
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.
Keywordscranial neural crest cells; disease models; loss-of-function analysis; palatogenesis; stage-matched gene expression analysis; temporal expression patterns
RightsThis article is licensed under a Creative Commons Attribution 4.0 International License.
URIhttps://repository.exst.jaxa.jp/dspace/handle/a-is/1399993


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