ArticleScientific reports2018
A Rapid CRISPR/Cas-based Mutagenesis Assay in Zebrafish for Identification of Genes Involved in Thyroid Morphogenesis and Function.
Article in Scientific reports, 2018. The graph could read no effect estimate from its abstract, so it casts no vote on the map. Cited by 27 papers.
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Who cites it
27 citing papers in PubMed, 60 citations in OpenAlex.
- The role of Nrf2 in thyroid maturation and hormone synthesis in vertebrate models.Life science alliance · 2026Article
- ift140 -Deficient Zebrafish as a Model for Kidney Cystogenesis and an F0-Based Screen for Genetic Modifiers of Kidney Cysts.Journal of the American Society of Nephrology : JASN · 2026Article
- Zebrafish: a key model for unraveling endocrine-disrupting effects on thyroid development and function.Frontiers in pharmacology · 2026Review
- Review
- A Novel Transgenic Model to Study Thyroid Axis Activity in Early Life Stage Medaka.Environmental science & technology · 2024Article
- Large-scale F0 CRISPR screens in vivo using MIC-Drop.Nature protocols · 2023Review
- Mesodermal FGF and BMP govern the sequential stages of zebrafish thyroid specification.Development (Cambridge, England) · 2023Article
- Lateral thinking in syndromic congenital cardiovascular disease.Disease models & mechanisms · 2023Article
- Duox is the primary NADPH oxidase responsible for ROS production during adult caudal fin regeneration in zebrafish.iScience · 2023Article
- Review
- Review
- Determining the Role of Maternally-Expressed Genes in Early Development with Maternal Crispants.Journal of visualized experiments : JoVE · 2021Article
- Identification of maternal-effect genes in zebrafish using maternal crispants.Development (Cambridge, England) · 2021Article
- Article
- Identification of the causative gene of a transparent phenotype of juvenile red sea bream Pagrus major.Heredity · 2021Article
- Synaptotagmin-Like Protein 2a Regulates Angiogenic Lumen Formation via Weibel-Palade Body Apical Secretion of Angiopoietin-2.Arteriosclerosis, thrombosis, and vascular biology · 2021Article
- Three-dimensional microscopy and image fusion reconstruction analysis of the thyroid gland during morphogenesis.FEBS open bio · 2021Article
- These Are the Genes You're Looking For: Finding Host Resistance Genes.Trends in microbiology · 2021Review
- CRISPR gRNA phenotypic screening in zebrafish reveals pro-regenerative genes in spinal cord injury.PLoS genetics · 2021Article
- Cre-Controlled CRISPR mutagenesis provides fast and easy conditional gene inactivation in zebrafish.Nature communications · 2021Article
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Authors and funding
10 authors at 2 institutions in 2 countries.
Funding
No grant is acknowledged in the PubMed record.
Abstract
The foregut endoderm gives rise to several organs including liver, pancreas, lung and thyroid with important roles in human physiology. Understanding which genes and signalling pathways regulate their development is crucial for understanding developmental disorders as well as diseases in adulthood. We exploited unique advantages of the zebrafish model to develop a rapid and scalable CRISPR/Cas-based mutagenesis strategy aiming at the identification of genes involved in morphogenesis and function of the thyroid. Core elements of the mutagenesis assay comprise bi-allelic gene invalidation in somatic mutants, a non-invasive monitoring of thyroid development in live transgenic fish, complementary analyses of thyroid function in fixed specimens and quantitative analyses of mutagenesis efficiency by Illumina sequencing of individual fish. We successfully validated our mutagenesis-phenotyping strategy in experiments targeting genes with known functions in early thyroid morphogenesis (pax2a, nkx2.4b) and thyroid functional differentiation (duox, duoxa, tshr). We also demonstrate that duox and duoxa crispants phenocopy thyroid phenotypes previously observed in human patients with bi-allelic DUOX2 and DUOXA2 mutations. The proposed combination of efficient mutagenesis protocols, rapid non-invasive phenotyping and sensitive genotyping holds great potential to systematically characterize the function of larger candidate gene panels during thyroid development and is applicable to other organs and tissues.
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