Disruption of fos causes craniofacial anomalies in developing zebrafish.

Autor:	Maili L; Department of Pediatrics, McGovern Medical School at the University of Texas Health Science Center at Houston, Houston, TX, United States.; Genetics and Epigenetics Graduate Program, The University of Texas MD Anderson Cancer Center UTHealth Houston Graduate School of Biomedical Sciences, Houston, TX, United States., Tandon B; Department of Pediatrics, McGovern Medical School at the University of Texas Health Science Center at Houston, Houston, TX, United States., Yuan Q; Department of Pediatrics, McGovern Medical School at the University of Texas Health Science Center at Houston, Houston, TX, United States., Menezes S; Center for Craniofacial Research, University of Texas Health Science Center School of Dentistry at Houston, Houston, TX, United States., Chiu F; Department of Pediatrics, McGovern Medical School at the University of Texas Health Science Center at Houston, Houston, TX, United States., Hashmi SS; Department of Pediatrics, McGovern Medical School at the University of Texas Health Science Center at Houston, Houston, TX, United States., Letra A; Center for Craniofacial Research, University of Texas Health Science Center School of Dentistry at Houston, Houston, TX, United States.; Department of Diagnostic and Biomedical Sciences, University of Texas Health Science Center School of Dentistry at Houston, Houston, TX, United States., Eisenhoffer GT; Genetics and Epigenetics Graduate Program, The University of Texas MD Anderson Cancer Center UTHealth Houston Graduate School of Biomedical Sciences, Houston, TX, United States.; Department of Genetics, The University of Texas MD Anderson Cancer Center, Houston, TX, United States., Hecht JT; Department of Pediatrics, McGovern Medical School at the University of Texas Health Science Center at Houston, Houston, TX, United States.; Genetics and Epigenetics Graduate Program, The University of Texas MD Anderson Cancer Center UTHealth Houston Graduate School of Biomedical Sciences, Houston, TX, United States.; Center for Craniofacial Research, University of Texas Health Science Center School of Dentistry at Houston, Houston, TX, United States.
Jazyk:	angličtina
Zdroj:	Frontiers in cell and developmental biology [Front Cell Dev Biol] 2023 Aug 16; Vol. 11, pp. 1141893. Date of Electronic Publication: 2023 Aug 16 (Print Publication: 2023).
DOI:	10.3389/fcell.2023.1141893
Abstrakt:	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. Competing Interests: The authors declare that the research was conducted in the absence of any commercial or financial relationships that could be construed as a potential conflict of interest. (Copyright © 2023 Maili, Tandon, Yuan, Menezes, Chiu, Hashmi, Letra, Eisenhoffer and Hecht.)
Databáze:	MEDLINE
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