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Subject collection: Zebrafish as a Disease Model

  • RESEARCH ARTICLE
    DnaJ-PKAc fusion induces liver inflammation in a zebrafish model of fibrolamellar carcinoma
    Sofia de Oliveira, Ruth A. Houseright, Benjamin G. Korte, Anna Huttenlocher
    Disease Models & Mechanisms 2020 13: dmm042564 doi: 10.1242/dmm.042564 Published 30 April 2020

    Summary: Inflammation and Caspase-a activation occur early in a new zebrafish model for fibrolamellar carcinoma (FLC). Pharmacological inhibition of TNFα secretion and caspase-1 might be useful approaches to treat FLC patients.

  • REVIEW
    Zebrafish as a tool to study schizophrenia-associated copy number variants
    Philip D. Campbell, Michael Granato
    Disease Models & Mechanisms 2020 13: dmm043877 doi: 10.1242/dmm.043877 Published 29 April 2020

    Summary: Specific copy number variants significantly increase the risk of schizophrenia, although their mechanisms are incompletely understood. We review the zebrafish as a tool to begin to decipher the complex biology of these rare variants.

  • EDITORIAL
    Casting a wide net: use of diverse model organisms to advance toxicology
    Mark E. Hahn, Kirsten C. Sadler
    Disease Models & Mechanisms 2020 13: dmm043844 doi: 10.1242/dmm.043844 Published 1 April 2020

    Summary: This Editorial highlights the value of utilizing evolutionarily diverse model systems, including zebrafish, in basic toxicology research to investigate mechanisms of chemical-induced disease.

  • REVIEW
    Zebrafish models of sarcopenia
    Alon Daya, Rajashekar Donaka, David Karasik
    Disease Models & Mechanisms 2020 13: dmm042689 doi: 10.1242/dmm.042689 Published 30 March 2020

    Summary: Zebrafish and other small fish have become powerful disease models. Here, we summarize the evidence for the utility of small teleost models for genetic research in sarcopenia – the age-related loss of muscle mass and function.

  • RESOURCE ARTICLE
    Establishment and validation of an endoplasmic reticulum stress reporter to monitor zebrafish ATF6 activity in development and disease
    Eric M. Clark, Hannah J. T. Nonarath, Jonathan R. Bostrom, Brian A. Link
    Disease Models & Mechanisms 2020 13: dmm041426 doi: 10.1242/dmm.041426 Published 28 January 2020

    Summary: In this study, we validate transgenic zebrafish generated to specifically report the activity of ATF6, representing a major branch of the endoplasmic reticulum stress pathway with functions in development and disease.

  • REVIEW
    Determining macrophage versus neutrophil contributions to innate immunity using larval zebrafish
    Emily E. Rosowski
    Disease Models & Mechanisms 2020 13: dmm041889 doi: 10.1242/dmm.041889 Published 9 January 2020

    Summary: Macrophages and neutrophils are distinct innate immune cells with diverse roles in diverse inflammatory contexts. Recent research in larval zebrafish using cell-specific depletion methods has revealed new insights into these cells' functions.

  • RESEARCH ARTICLE
    A model for reticular dysgenesis shows impaired sensory organ development and hair cell regeneration linked to cellular stress
    Alberto Rissone, Erin Jimenez, Kevin Bishop, Blake Carrington, Claire Slevin, Stephen M. Wincovitch, Raman Sood, Fabio Candotti, Shawn M. Burgess
    Disease Models & Mechanisms 2019 12: dmm040170 doi: 10.1242/dmm.040170 Published 20 December 2019

    Summary: A zebrafish model of reticular dysgenesis reveals hair cell developmental deficits that can be partially rescued by antioxidants, pointing to their potential use as a therapeutic treatment for reticular dysgenesis patients.

  • RESEARCH ARTICLE
    Perturbation of the titin/MURF1 signaling complex is associated with hypertrophic cardiomyopathy in a fish model and in human patients
    Yuta Higashikuse, Nishant Mittal, Takuro Arimura, Sung Han Yoon, Mayumi Oda, Hirokazu Enomoto, Ruri Kaneda, Fumiyuki Hattori, Takeshi Suzuki, Atsushi Kawakami, Alexander Gasch, Tetsushi Furukawa, Siegfried Labeit, Keiichi Fukuda, Akinori Kimura, Shinji Makino
    Disease Models & Mechanisms 2019 12: dmm041103 doi: 10.1242/dmm.041103 Published 15 November 2019

    Summary: The authors identified and characterized a medaka mutation in titin that leads to a phenotype similar to hypertrophic cardiomyopathy. Similar mutations were also observed in human patients.

  • RESEARCH ARTICLE
    Targeted knockout of GABA-A receptor gamma 2 subunit provokes transient light-induced reflex seizures in zebrafish larvae
    Meijiang Liao, Uday Kundap, Richard E. Rosch, Dominic R. W. Burrows, Martin P. Meyer, Bouchra Ouled Amar Bencheikh, Patrick Cossette, Éric Samarut
    Disease Models & Mechanisms 2019 12: dmm040782 doi: 10.1242/dmm.040782 Published 11 November 2019

    Summary: The authors present a novel in vivo genetic model of idiopathic epilepsy in zebrafish (gabrg2−/−) to aid the study of ictogenesis and provide a convenient genetic tool for drug screening.

  • RESOURCE ARTICLE
    Deep learning enables automated volumetric assessments of cardiac function in zebrafish
    Alexander A. Akerberg, Caroline E. Burns, C. Geoffrey Burns, Christopher Nguyen
    Disease Models & Mechanisms 2019 12: dmm040188 doi: 10.1242/dmm.040188 Published 25 October 2019

    Summary: The authors present CFIN, a deep learning-based image-analysis platform to automatically analyze dynamic light-sheet fluorescence microscopy images and determine volumetric indices of cardiac function in embryonic zebrafish.

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