The developing utility of zebrafish models of neurological and neuropsychiatric disorders: A critical review

BD Fontana, NJ Mezzomo, AV Kalueff… - Experimental …, 2018 - Elsevier
Zebrafish (Danio rerio) have become a powerful tool in neuroscience research due to their
genetic tractability, molecular/physiological conservation, small body size, ease of …

Zebrafish models of neurodevelopmental disorders: past, present, and future

C Sakai, S Ijaz, EJ Hoffman - Frontiers in molecular neuroscience, 2018 - frontiersin.org
Zebrafish are increasingly being utilized as a model system to investigate the function of the
growing list of risk genes associated with neurodevelopmental disorders. This is due in large …

Zebrafish models in neuropsychopharmacology and CNS drug discovery

KM Khan, AD Collier, DA Meshalkina… - British journal of …, 2017 - Wiley Online Library
Despite the high prevalence of neuropsychiatric disorders, their aetiology and molecular
mechanisms remain poorly understood. The zebrafish (Danio rerio) is increasingly utilized …

STXBP1 encephalopathies: clinical spectrum, disease mechanisms, and therapeutic strategies

D Abramov, NGL Guiberson… - Journal of …, 2021 - Wiley Online Library
Abstract Mutations in Munc18‐1/STXBP1 (syntaxin‐binding protein 1) are linked to various
severe early epileptic encephalopathies and neurodevelopmental disorders. Heterozygous …

Using zebrafish for investigating the molecular mechanisms of drug‐induced cardiotoxicity

ZZ Zakaria, FM Benslimane… - BioMed research …, 2018 - Wiley Online Library
Over the last decade, the zebrafish (Danio rerio) has emerged as a model organism for
cardiovascular research. Zebrafish have several advantages over mammalian models. For …

[HTML][HTML] Seizing the moment: Zebrafish epilepsy models

K Gawel, M Langlois, T Martins, W van der Ent… - Neuroscience & …, 2020 - Elsevier
Zebrafish are now widely accepted as a valuable animal model for a number of different
central nervous system (CNS) diseases. They are suitable both for elucidating the origin of …

Protein instability, haploinsufficiency, and cortical hyper-excitability underlie STXBP1 encephalopathy

J Kovačević, G Maroteaux, D Schut, M Loos, M Dubey… - Brain, 2018 - academic.oup.com
De novo heterozygous mutations in STXBP1/Munc18-1 cause early infantile epileptic
encephalopathies (EIEE4, OMIM# 612164) characterized by infantile epilepsy …

Phenotypic analysis of catastrophic childhood epilepsy genes

A Griffin, C Carpenter, J Liu, R Paterno, B Grone… - Communications …, 2021 - nature.com
Genetic engineering techniques have contributed to the now widespread use of zebrafish to
investigate gene function, but zebrafish-based human disease studies, and particularly for …

Pyridoxine-dependent epilepsy in zebrafish caused by Aldh7a1 deficiency

IA Pena, Y Roussel, K Daniel, K Mongeon… - Genetics, 2017 - academic.oup.com
Pyridoxine-dependent epilepsy (PDE) is a severe neonatal seizure disorder and is here
modeled in aldh7a1-/-zebrafish. Mutant larvae display spontaneous.. Pyridoxine-dependent …

Mechanism-based rescue of Munc18-1 dysfunction in varied encephalopathies by chemical chaperones

NGL Guiberson, A Pineda, D Abramov, P Kharel… - Nature …, 2018 - nature.com
Heterozygous de novo mutations in the neuronal protein Munc18-1 are linked to epilepsies,
intellectual disability, movement disorders, and neurodegeneration. These devastating …