RRC ID 83773
著者 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.
タイトル Expression analysis of genes including Zfhx4 in mice and zebrafish reveals a temporospatial conserved molecular basis underlying craniofacial development.
ジャーナル Dev Dyn
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.
巻・号 254(3)
ページ 257-271
公開日 2025-3-1
DOI 10.1002/dvdy.740
PMID 39320016
PMC PMC11877995
MeSH Animals Craniofacial Abnormalities / genetics Gene Expression Regulation, Developmental* Homeodomain Proteins* / genetics Homeodomain Proteins* / metabolism Mice Skull* / embryology Skull* / metabolism Zebrafish* / embryology Zebrafish* / genetics Zebrafish Proteins* / genetics Zebrafish Proteins* / metabolism
IF 3.275
リソース情報
ゼブラフィッシュ RIKEN WT (RW)