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Zebrafish Models of Craniofacial Malformations: Interactions of Environmental Factors.

ABSTRACT: The zebrafish is an appealing model organism for investigating the genetic (G) and environmental (E) factors, as well as their interactions (GxE), which contribute to craniofacial malformations. Here, we review zebrafish studies on environmental factors involved in the etiology of craniofacial malformations in humans including maternal smoking, alcohol consumption, nutrition and drug use. As an example, we focus on the (cleft) palate, for which the zebrafish ethmoid plate is a good model. This review highlights the importance of investigating ExE interactions and discusses the variable effects of exposure to environmental factors on craniofacial development depending on dosage, exposure time and developmental stage. Zebrafish also promise to be a good tool to study novel craniofacial teratogens and toxin mixtures. Lastly, we discuss the handful of studies on gene-alcohol interactions using mutant sensitivity screens and reverse genetic techniques. We expect that studies addressing complex interactions (ExE and GxE) in craniofacial malformations will increase in the coming years. These are likely to uncover currently unknown mechanisms with implications for the prevention of craniofacial malformations. The zebrafish appears to be an excellent complementary model with high translational value to study these complex interactions.

SUBMITTER: Raterman ST

PROVIDER: S-EPMC7701217 | biostudies-literature |

REPOSITORIES: biostudies-literature

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Project description:Current standards for husbandry and maintenance of zebrafish and other aquatic species in the laboratory are diverse, and are subject to laboratory performance, engineering, and practice standards (the Guide), institutional interpretation, national animal welfare laws, and cultural differences. Consequently, it is difficult, and probably not advantageous, to establish a single standard in view of the hardy nature of zebrafish and the diversity of research requirements it is used to address. Based on their natural habitat, zebrafish can thrive in a variety of environmental conditions, which is a specific advantage for working with this laboratory organism. However, it also makes reporting and reproducibility difficult, because variations in the husbandry and environmental conditions, including the environmental conditions before and during experiments, are often underreported in the scientific literature. This lack of consistency presents a potential problem for research reproducibility. To begin addressing this emerging scientific gap, the National Institutes of Health's (NIH) Office of Research Infrastructure Programs (ORIP), Division of Construction and Instruments (DCI), hosted a workshop in late 2017, entitled "Zebrafish and Other Aquatic Models: Reporting of Environmental Husbandry Conditions for Rigorous Experiments and Reproducible Results," that was attended by ∼60 participants. The objectives of the workshop were to bring together a diverse group of stakeholders-researchers, facility managers, veterinarians, journal editors, commercial vendors, and others to (1) review current husbandry and environmental management practices for the care of zebrafish and other aquatic organisms in the laboratory and to (2) propose a process for the development of a minimal set of environmental parameters that should be reported in publications to ensure rigor and robustness of experiments and reproducible outcomes. The participants also discussed how these recommendations, as an initial step, might be collected, disseminated, implemented, and improved upon after future iteration.

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Zebrafish Models of Craniofacial Malformations: Interactions of Environmental Factors.

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