Integrating non-mammalian model organisms in the diagnosis of rare genetic diseases in humans

S Yamamoto, O Kanca, MF Wangler… - Nature Reviews …, 2024 - nature.com
Next-generation sequencing technology has rapidly accelerated the discovery of genetic
variants of interest in individuals with rare diseases. However, showing that these variants …

Hooked! Modeling human disease in zebrafish

C Santoriello, LI Zon - The Journal of clinical investigation, 2012 - Am Soc Clin Investig
Zebrafish have been widely used as a model system for studying developmental processes,
but in the last decade, they have also emerged as a valuable system for modeling human …

In vivo drug discovery in the zebrafish

LI Zon, RT Peterson - Nature reviews Drug discovery, 2005 - nature.com
The zebrafish has become a widely used model organism because of its fecundity, its
morphological and physiological similarity to mammals, the existence of many genomic tools …

Zebrafish behavior: opportunities and challenges

MB Orger, GG de Polavieja - Annual review of neuroscience, 2017 - annualreviews.org
A great challenge in neuroscience is understanding how activity in the brain gives rise to
behavior. The zebrafish is an ideal vertebrate model to address this challenge, thanks to the …

Drinks like a fish: zebra fish (Danio rerio) as a behavior genetic model to study alcohol effects

R Gerlai, M Lahav, S Guo, A Rosenthal - Pharmacology biochemistry and …, 2000 - Elsevier
Zebra fish may be an ideal vertebrate model system for numerous human diseases with
which the genetics and biological mechanisms of the disease may be studied. Zebra fish …

[HTML][HTML] Whole-brain activity maps reveal stereotyped, distributed networks for visuomotor behavior

R Portugues, CE Feierstein, F Engert, MB Orger - Neuron, 2014 - cell.com
Most behaviors, even simple innate reflexes, are mediated by circuits of neurons spanning
areas throughout the brain. However, in most cases, the distribution and dynamics of firing …

Zebrafish: an emerging model system for human disease and drug discovery

G Kari, U Rodeck, AP Dicker - Clinical Pharmacology & …, 2007 - Wiley Online Library
In vivo studies represent an essential step in drug development and currently rely largely on
mice, yet limitations of mammalian models motivated the search for complementary …

A brainstem integrator for self-location memory and positional homeostasis in zebrafish

E Yang, MF Zwart, B James, M Rubinov, Z Wei… - Cell, 2022 - cell.com
To track and control self-location, animals integrate their movements through space.
Representations of self-location are observed in the mammalian hippocampal formation, but …

The state of the art of the zebrafish model for toxicology and toxicologic pathology research—advantages and current limitations

JM Spitsbergen, ML Kent - Toxicologic pathology, 2003 - journals.sagepub.com
The zebrafish (Danio rerio) is now the pre-eminent vertebrate model system for clarification
of the roles of specific genes and signaling pathways in development. The zebrafish …

The art and design of genetic screens: zebrafish

EE Patton, LI Zon - Nature Reviews Genetics, 2001 - nature.com
Inventive genetic screens in zebrafish are revealing new genetic pathways that control
vertebrate development, disease and behaviour. By exploiting the versatility of zebrafish …