[HTML][HTML] Drug screening in Scn1a zebrafish mutant identifies clemizole as a potential Dravet syndrome treatment

SC Baraban, MT Dinday, GA Hortopan - Nature communications, 2013 - nature.com
Dravet syndrome is a catastrophic pediatric epilepsy with severe intellectual disability, impaired
social development and persistent drug-resistant seizures. One of its primary monogenic …

Zebrafish as a model for studying genetic aspects of epilepsy

GA Hortopan, MT Dinday… - Disease models & …, 2010 - journals.biologists.com
Despite a long tradition of using rats and mice to model epilepsy, several aspects of rodent
biology limit their use in large-scale genetic and therapeutic drug screening programs. …

A large‐scale mutagenesis screen to identify seizure‐resistant zebrafish

SC Baraban, MT Dinday, PA Castro, S Chege… - …, 2007 - Wiley Online Library
Methods: Seizures were induced with pentylenetetrazole (PTZ). Zebrafish were analyzed
between 3 and 7 days postfertilization (dpf). Genome mutations were induced in founders by …

Large-scale phenotype-based antiepileptic drug screening in a zebrafish model of Dravet syndrome

MT Dinday, SC Baraban - Eneuro, 2015 - eneuro.org
Mutations in a voltage-gated sodium channel (SCN1A) result in Dravet Syndrome (DS), a
catastrophic childhood epilepsy. Zebrafish with a mutation in scn1Lab recapitulate salient …

Spontaneous seizures and altered gene expression in GABA signaling pathways in a mind bomb mutant zebrafish

GA Hortopan, MT Dinday, SC Baraban - Journal of Neuroscience, 2010 - Soc Neuroscience
Disruption of E3 ubiquitin ligase activity in immature zebrafish mind bomb mutants leads to
a failure in Notch signaling, excessive numbers of neurons, and depletion of neural …

[HTML][HTML] Phenotypic analysis of catastrophic childhood epilepsy genes

…, B Grone, K Hamling, M Moog, MT Dinday… - 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 …

Altered glycolysis and mitochondrial respiration in a zebrafish model of Dravet syndrome

MG Kumar, S Rowley, R Fulton, MT Dinday… - ENeuro, 2016 - eneuro.org
Altered metabolism is an important feature of many epileptic syndromes but has not been
reported in Dravet syndrome (DS), a catastrophic childhood epilepsy associated with …

Expression and function of KCNQ channels in larval zebrafish

SW Chege, GA Hortopan, M T. Dinday… - Developmental …, 2012 - Wiley Online Library
Members of the K v 7 family generate a subthreshold potassium current, termed M‐current,
that regulates the excitability of principal central neurons. Mutations in two members of this …

Enhancing glucose metabolism via gluconeogenesis is therapeutic in a zebrafish model of Dravet syndrome

R Banerji, C Huynh, F Figueroa, MT Dinday… - Brain …, 2021 - academic.oup.com
Energy-producing pathways are novel therapeutic targets for the treatment of neurodevelopmental
disorders. Here, we focussed on correcting metabolic defects in a catastrophic …

Abnormal cortical cells and astrocytomas in the Eker rat model of tuberous sclerosis complex

DK Takahashi, MT Dinday, NM Barbaro… - Epilepsia, 2004 - Wiley Online Library
Purpose: In patients with tuberous sclerosis complex (TSC), a wide range of neurologic
abnormalities develop, including mental retardation and seizures. Brains from TSC patients are …