Fetal alcohol spectrum disorder (FASD) describes a wide range of structural deficits and cognitive impairments. FASD impacts up to 5% of children born in the United States each year, making ethanol one of the most common teratogens. Due to limitations and ethical concerns, studies in humans are limited in their ability to study FASD. Animal models have proven critical in identifying and characterizing the mechanisms underlying FASD. In this review, we will focus on the attributes of zebrafish that make it a strong model in which to study ethanol-induced developmental defects. Zebrafish have several attributes that make it an ideal model in which to study FASD. Zebrafish produced large numbers of externally fertilized, translucent embryos. With a high degree of genetic amenability, zebrafish are at the forefront of identifying and characterizing the gene-ethanol interactions that underlie FASD. Work from multiple labs has shown that embryonic ethanol exposures result in defects in craniofacial, cardiac, ocular, and neural development. In addition to structural defects, ethanol-induced cognitive and behavioral impairments have been studied in zebrafish. Building upon these studies, work has identified ethanol-sensitive loci that underlie the developmental defects. However, analyses show there is still much to be learned of these gene-ethanol interactions. The zebrafish is ideally suited to expand our understanding of gene-ethanol interactions and their impact on FASD. Because of the conservation of gene function between zebrafish and humans, these studies will directly translate to studies of candidate genes in human populations and allow for better diagnosis and treatment of FASD.
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http://dx.doi.org/10.1002/dvg.23460 | DOI Listing |
Unlabelled: Fetal Alcohol Spectrum Disorders (FASD) describes a wide array of neurological defects and craniofacial malformations, associated with ethanol teratogenicity. While there is growing evidence for a genetic component to FASD, little is known of the genes underlying these ethanol-induced defects. Along with timing and dosage, genetic predispositions may help explain the variability within FASD.
View Article and Find Full Text PDFReprod Toxicol
January 2025
University of Louisville, School of Medicine, Department of Biochemistry and Molecular Genetics, 319 Abraham Flexner Way, Louisville, KY 40202, USA. Electronic address:
Fetal Alcohol Spectrum Disorders (FASD) describes a wide array of neurological defects and craniofacial malformations, associated with ethanol teratogenicity. While there is growing evidence for a genetic component to FASD, little is known of the genes underlying these ethanol-induced defects. Along with timing and dosage, genetic predispositions may help explain the variability within FASD.
View Article and Find Full Text PDFDev Dyn
March 2025
Department of Dentistry-Orthodontics and Craniofacial Biology, Research Institute for Medical Innovation, Radboud University Medical Center, Nijmegen, The Netherlands.
Background: FOXE1 mutations in humans are associated with cleft palate and hypothyroidism. We previously developed a foxe1 mutant zebrafish demonstrating mineralization defects in larvae. In the present study, we investigate the thyroid status and skeletal phenotype of adult foxe1 mutants.
View Article and Find Full Text PDFGenesis
November 2021
Department of Biochemistry and Molecular Genetics, Alcohol Research Center, University of Louisville, Louisville, Kentucky, USA.
Fetal alcohol spectrum disorder (FASD) describes a wide range of structural deficits and cognitive impairments. FASD impacts up to 5% of children born in the United States each year, making ethanol one of the most common teratogens. Due to limitations and ethical concerns, studies in humans are limited in their ability to study FASD.
View Article and Find Full Text PDFReprod Toxicol
October 2021
Bowles Center on Alcohol Studies, University of North Carolina, Chapel Hill, NC, USA; Department of Cell Biology and Physiology, University of North Carolina, Chapel Hill, NC, USA. Electronic address:
Neurulation-stage alcohol exposure (NAE; embryonic day [E] 8-10) is associated with midline craniofacial and CNS defects that likely arise from disruption of morphogen pathways, such as Sonic hedgehog (Shh). Notably, midline anomalies are also a hallmark of genetic ciliopathies such as Joubert syndrome. We tested whether NAE alters Shh pathway signaling and the number and function of primary cilia, organelles critical for Shh pathway transduction.
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