PUBLICATION

Disruption of fos causes craniofacial anomalies in developing zebrafish

Authors
Maili, L., Tandon, B., Yuan, Q., Menezes, S., Chiu, F., Hashmi, S.S., Letra, A., Eisenhoffer, G.T., Hecht, J.T.
ID
ZDB-PUB-230904-62
Date
2023
Source
Frontiers in cell and developmental biology   11: 11418931141893 (Journal)
Registered Authors
Eisenhoffer, George
Keywords
craniofacial development, fos, geometric morphometrics, morphogenesis, orofacial cleft
MeSH Terms
none
PubMed
37664458 Full text @ Front Cell Dev Biol
Abstract
Craniofacial development is a complex and tightly regulated process and disruptions can lead to structural birth defects, the most common being nonsyndromic cleft lip and palate (NSCLP). Previously, we identified FOS as a candidate regulator of NSCLP through family-based association studies, yet its specific contributions to oral and palatal formation are poorly understood. This study investigated the role of fos during zebrafish craniofacial development through genetic disruption and knockdown approaches. Fos was expressed in the periderm, olfactory epithelium and other cell populations in the head. Genetic perturbation of fos produced an abnormal craniofacial phenotype with a hypoplastic oral cavity that showed significant changes in midface dimensions by quantitative facial morphometric analysis. Loss and knockdown of fos caused increased cell apoptosis in the head, followed by a significant reduction in cranial neural crest cells (CNCCs) populating the upper and lower jaws. These changes resulted in abnormalities of cartilage, bone and pharyngeal teeth formation. Periderm cells surrounding the oral cavity showed altered morphology and a subset of cells in the upper and lower lip showed disrupted Wnt/β-catenin activation, consistent with modified inductive interactions between mesenchymal and epithelial cells. Taken together, these findings demonstrate that perturbation of fos has detrimental effects on oral epithelial and CNCC-derived tissues suggesting that it plays a critical role in zebrafish craniofacial development and a potential role in NSCLP.
Genes / Markers
Marker Marker Type Name
fosaaGENEv-fos FBJ murine osteosarcoma viral oncogene homolog Aa
fosabGENEv-fos FBJ murine osteosarcoma viral oncogene homolog Ab
fosbGENEFBJ murine osteosarcoma viral oncogene homolog B
fosl1aGENEFOS like 1, AP-1 transcription factor subunit a
fosl1bGENEFOS like 1, AP-1 transcription factor subunit b
fosl2GENEFOS like 2, AP-1 transcription factor subunit
tp53GENEtumor protein p53
tyrGENEtyrosinase
1 - 8 of 8
Show
Figures
Figure Gallery (6 images)
Show all Figures
Expression
Phenotype
Fish Conditions Stage Phenotype Figure
WT + CRISPR1-fosab + CRISPR2-fosabstandard conditionsPrim-5
WT + CRISPR1-fosab + CRISPR2-fosabstandard conditionsPrim-5
WT + CRISPR1-fosab + CRISPR2-fosabstandard conditionsLong-pec
WT + CRISPR1-fosab + CRISPR2-fosabstandard conditionsProtruding-mouth
WT + CRISPR1-fosab + CRISPR2-fosabstandard conditionsDay 5
WT + CRISPR1-fosab + CRISPR2-fosabstandard conditionsDay 5
WT + CRISPR1-fosab + CRISPR2-fosabstandard conditionsDay 5
WT + CRISPR1-fosab + CRISPR2-fosabstandard conditionsDay 5
WT + CRISPR1-fosab + CRISPR2-fosabstandard conditionsDay 5
WT + CRISPR1-fosab + CRISPR2-fosabstandard conditionsDay 5
1 - 10 of 93
Show
Mutations / Transgenics
Allele Construct Type Affected Genomic Region
ba2TgTransgenic Insertion
    gz7TgTransgenic Insertion
      ia4TgTransgenic Insertion
        1 - 3 of 3
        Show
        Human Disease / Model
        Human Disease Fish Conditions Evidence
        orofacial cleftTAS
        1 - 1 of 1
        Show
        Sequence Targeting Reagents
        Target Reagent Reagent Type
        fosabCRISPR1-fosabCRISPR
        fosabCRISPR2-fosabCRISPR
        fosabMO2-fosabMRPHLNO
        tp53MO4-tp53MRPHLNO
        tyrCRISPR1-tyrCRISPR
        1 - 5 of 5
        Show
        Fish
        Fish
        ba2Tg + CRISPR1-fosab + CRISPR2-fosab
        ba2Tg + MO2-fosab
        gz7Tg + CRISPR1-fosab + CRISPR2-fosab
        gz7Tg + MO2-fosab
        WT
        WT + CRISPR1-fosab + CRISPR2-fosab
        WT + MO2-fosab
        1 - 7 of 7
        Show
        Antibodies
        Orthology
        Engineered Foreign Genes
        Marker Marker Type Name
        EGFPEFGEGFP
        GFPEFGGFP
        1 - 2 of 2
        Show
        Mapping
        Your Input Welcome
        We welcome your input and comments. Please use this form to recommend updates to the information in ZFIN. We appreciate as much detail as possible and references as appropriate. We will review your comments promptly.
        Citation
        Maili, L., Tandon, B., Yuan, Q., Menezes, S., Chiu, F., Hashmi, S.S., Letra, A., Eisenhoffer, G.T., Hecht, J.T. (2023) Disruption of fos causes craniofacial anomalies in developing zebrafish. Frontiers in cell and developmental biology. 11:11418931141893.