Facial analytics based on a coordinate extrapolation system (zFACE) for morphometric phenotyping of developing zebrafish.

Autor:	Maili L; Pediatric Research Center, Department of Pediatrics, McGovern Medical School University of Texas Health Science Center-Houston, Houston, TX 77030, USA.; Genetics and Epigenetics Graduate Program, The University of Texas MD Anderson Cancer Center UTHealth Houston Graduate School of Biomedical Sciences, Houston, TX 77030, USA., Ruiz OE; Department of Genetics, The University of Texas MD Anderson Cancer Center, Houston, TX 77030, USA., Kahan PH; Department of Genetics, The University of Texas MD Anderson Cancer Center, Houston, TX 77030, USA., Chiu F; Pediatric Research Center, Department of Pediatrics, McGovern Medical School University of Texas Health Science Center-Houston, Houston, TX 77030, USA., Larson ST; Department of Genetics, The University of Texas MD Anderson Cancer Center, Houston, TX 77030, USA., Hashmi SS; Pediatric Research Center, Department of Pediatrics, McGovern Medical School University of Texas Health Science Center-Houston, Houston, TX 77030, USA., Hecht JT; Pediatric Research Center, Department of Pediatrics, McGovern Medical School University of Texas Health Science Center-Houston, Houston, TX 77030, USA.; Genetics and Epigenetics Graduate Program, The University of Texas MD Anderson Cancer Center UTHealth Houston Graduate School of Biomedical Sciences, Houston, TX 77030, USA., Eisenhoffer GT; Genetics and Epigenetics Graduate Program, The University of Texas MD Anderson Cancer Center UTHealth Houston Graduate School of Biomedical Sciences, Houston, TX 77030, USA.; Department of Genetics, The University of Texas MD Anderson Cancer Center, Houston, TX 77030, USA.
Jazyk:	angličtina
Zdroj:	Disease models & mechanisms [Dis Model Mech] 2023 Jun 01; Vol. 16 (6). Date of Electronic Publication: 2023 Jun 02.
DOI:	10.1242/dmm.049868
Abstrakt:	Facial development requires a complex and coordinated series of cellular events that, when perturbed, can lead to structural birth defects. A quantitative approach to quickly assess morphological changes could address how genetic or environmental inputs lead to differences in facial shape and promote malformations. Here, we report on a method to rapidly analyze craniofacial development in zebrafish embryos using facial analytics based on a coordinate extrapolation system, termed zFACE. Confocal images capture facial structures and morphometric data are quantified based on anatomical landmarks present during development. The quantitative morphometric data can detect phenotypic variation and inform on changes in facial morphology. We applied this approach to show that loss of smarca4a in developing zebrafish leads to craniofacial anomalies, microcephaly and alterations in brain morphology. These changes are characteristic of Coffin-Siris syndrome, a rare human genetic disorder associated with mutations in SMARCA4. Multivariate analysis of zFACE data facilitated the classification of smarca4a mutants based on changes in specific phenotypic characteristics. Together, zFACE provides a way to rapidly and quantitatively assess the impact of genetic alterations on craniofacial development in zebrafish. Competing Interests: Competing interests The authors declare no competing or financial interests. (© 2023. Published by The Company of Biologists Ltd.)
Databáze:	MEDLINE
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