An - Regulatory Axis Controls Midface Morphogenesis in Vertebrates

Overview

Journal Development

Specialty Biology

Date 2020 Nov 25

PMID 33234718

Citations 12

Authors

Shannon H Carroll

Claudio Macias Trevino

Edward B Li

Kenta Kawasaki

Nikita Myers

Shawn A Hallett

Nora Alhazmi

Justin Cotney

Russ P Carstens

Eric C Liao

Affiliations

Soon will be listed here.

Abstract

and are important for palate development across vertebrates. In zebrafish, we found that regulates the expression of We detailed overlapping and expression in mouse orofacial epithelium. In zebrafish, and share expression in periderm, frontonasal ectoderm and oral epithelium. Genetic disruption of and in zebrafish resulted in cleft of the anterior neurocranium. The mutant also developed cleft of the mouth opening. Lineage tracing of cranial neural crest cells revealed that the cleft resulted not from migration defect, but from impaired chondrogenesis. Analysis of aberrant cells within the cleft revealed expression of , and , and these cells were adjacent to and cells. Breeding of mouse ; ; compound mutants suggested genetic interaction, as the triple homozygote and the ; double homozygote were not observed. Further, heterozygosity reduced cleft severity. These studies highlight the complementary analysis of and in mouse and zebrafish, and identify a unique aberrant cell population in zebrafish expressing , and Future work characterizing this cell population will yield additional insight into cleft pathogenesis.

Citing Articles

PRMT1-methylated MSX1 phase separates to control palate development.

Meng L, Jiang Y, You J, Chen Y, Guo S, Chen L Nat Commun. 2025; 16(1):949.

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Genetic requirement of to regulate noncanonical Wnt signaling and during embryonic convergent extension and craniofacial morphogenesis.

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Expression analysis of genes including Zfhx4 in mice and zebrafish reveals a temporospatial conserved molecular basis underlying craniofacial development.

Liu S, Xu L, Kashima M, Narumi R, Takahata Y, Nakamura E Dev Dyn. 2024; 254(3):257-271.

PMID: 39320016 PMC: 11877995. DOI: 10.1002/dvdy.740.

Functional analysis of ESRP1/2 gene variants and CTNND1 isoforms in orofacial cleft pathogenesis.

Caetano da Silva C, Macias Trevino C, Mitchell J, Murali H, Tsimbal C, Dalessandro E Commun Biol. 2024; 7(1):1040.

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Functional analysis of gene variants and isoforms in orofacial cleft pathogenesis.

Caetano da Silva C, Macias Trevino C, Mitchell J, Murali H, Tsimbal C, Dalessandro E bioRxiv. 2024; .

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