A Manual Small Molecule Screen Approaching High-throughput Using Zebrafish Embryos

Poureetezadi, Shahram Jevin; Donahue, Eric K.; Wingert, Rebecca A.

doi:10.3791/52063-v

Cited by 2 publications

(1 citation statement)

References 25 publications

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“…Zebrafish also possess a high fecundity, allowing for genetic screens [ 134 , 135 , 136 , 137 , 138 , 139 , 140 , 141 , 142 , 143 , 144 ] and high throughput assays to assess varied compounds and their role in developmental processes [ 145 , 146 , 147 , 148 , 149 , 150 , 151 , 152 ]. For example, RAR and RXR agonists/antagonists and their role in developmental processes can be explored by adding these compounds at various times ( Figure 2 ).…”

Section: Ra and Zebrafish A Backgroundmentioning

confidence: 99%

Zebrafish as a Model to Study Retinoic Acid Signaling in Development and Disease

Hawkins

Wingert

2023

Biomedicines

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Retinoic acid (RA) is a metabolite of vitamin A (retinol) that plays various roles in development to influence differentiation, patterning, and organogenesis. RA also serves as a crucial homeostatic regulator in adult tissues. The role of RA and its associated pathways are well conserved from zebrafish to humans in both development and disease. This makes the zebrafish a natural model for further interrogation into the functions of RA and RA-associated maladies for the sake of basic research, as well as human health. In this review, we explore both foundational and recent studies using zebrafish as a translational model for investigating RA from the molecular to the organismal scale.

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Section: Ra and Zebrafish A Backgroundmentioning

confidence: 99%

Zebrafish as a Model to Study Retinoic Acid Signaling in Development and Disease

Hawkins

Wingert

2023

Biomedicines

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Zebrafish models for human ALA-dehydratase-deficient porphyria (ADP) and hereditary coproporphyria (HCP) generated with TALEN and CRISPR-Cas9

Zhang¹,

Meng²,

Niu³

et al. 2017

Preprint

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Running title: Zebrafish models for human porphyrias ADP and HCP 21 22 23 24 42 provide invaluable resources for elucidating novel pathological aspects of porphyrias, 43 evaluating mutated forms of human ALAD and CPOX, discovering new therapeutic 44 targets and developing effective drugs for these complex genetic diseases. Our studies 45 3 also highlight generation of zebrafish models for human diseases with two versatile 46 genome-editing tools. 47 48 49 50 51 4 157 6-bp deletion resulting in deletion of 2 amino acids (ZFIN allele nomenclature 158 designation, cpox sus004 ) in a highly conserved region (Hanaoka et al., 2006) and the 159 Brownlie, A., Donovan, A., Pratt, S. J. , Paw, B. H., Oates, A. C., Brugnara, C., Witkowska, H. 515 25 E., Sassa, S. and Zon, L. I. (1998). Positional cloning of the zebrafish sauternes gene: a model for 516 congenital sideroblastic anaemia. Nat Genet 20, 244-50. 517 Cappellini, M. D., Brancaleoni, V., Graziadei, G., Tavazzi, D. and Di Pierro, E. (2010). 518Porphyrias at a glance: diagnosis and treatment. Intern Emerg Med 5 Suppl 1, S73-80. 519 Chiabrando, D., Mercurio, S. and Tolosano, E. (2014). Heme and erythropoieis: more than a 520 structural role. Haematologica 99, 973-83. 521 Childs , S., Weinstein, B. M., Mohideen, M. A., Donohue, S., Bonkovsky, H. and Fishman, M. 522 C. (2000). Zebrafish dracula encodes ferrochelatase and its mutation provides a model for erythropoietic 523 protoporphyria. 527 5-aminolevulinic acid in blood plasma, tissues and cell cultures by high-performance liquid 528 chromatography with electrochemical detection. J Chromatogr B Biomed Sci Appl 695, 245-50. 529

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A Manual Small Molecule Screen Approaching High-throughput Using Zebrafish Embryos

Cited by 2 publications

References 25 publications

Zebrafish as a Model to Study Retinoic Acid Signaling in Development and Disease

Zebrafish as a Model to Study Retinoic Acid Signaling in Development and Disease

Zebrafish models for human ALA-dehydratase-deficient porphyria (ADP) and hereditary coproporphyria (HCP) generated with TALEN and CRISPR-Cas9

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