Swimming against ALS: How to model disease in zebrafish for pathophysiological and behavioral studies

Integrative genetic analysis illuminates ALS heritability and identifies risk genes

(2023) Salim Megat et al.   Nature Communications

Human stem cell models of neurodegeneration: From basic science of amyotrophic lateral sclerosis to clinical translation

(2022) Elisa Giacomelli et al.   Cell Stem Cell

The Cyanotoxin 2,4-DAB Reduces Viability and Causes Behavioral and Molecular Dysfunctions Associated with Neurodegeneration in Larval Zebrafish

(2022) Rubia M. Martin et al.   NEUROTOXICITY RESEARCH

Recent Updates on the Genetics of Amyotrophic Lateral Sclerosis and Frontotemporal Dementia

(2022) Laxmi Kirola et al.   MOLECULAR NEUROBIOLOGY

Modeling neurodegenerative disorders in zebrafish

(2022) Alim A. Bashirzade et al.   NEUROSCIENCE AND BIOBEHAVIORAL REVIEWS

Disruptions of circadian rhythms, sleep, and stress responses in zebrafish: New infrared-based activity monitoring assays for toxicity assessment

(2022) Rui F.O. Silva et al.   CHEMOSPHERE

Amyotrophic Lateral Sclerosis-Associated Persistent Organic Pollutant cis-Chlordane Causes GABAA-Independent Toxicity to Motor Neurons, Providing Evidence toward an Environmental Component of Sporadic Amyotrophic Lateral Sclerosis

(2022) Daniel Kulick et al.   ACS Chemical Neuroscience

Prematurely terminated intron-retaining mRNAs invade axons in SFPQ null-driven neurodegeneration and are a hallmark of ALS

(2022) Richard Taylor et al.   Nature Communications

Is Exposure to BMAA a Risk Factor for Neurodegenerative Diseases? A Response to a Critical Review of the BMAA Hypothesis

(2021) Dunlop RA et al.   NEUROTOXICITY RESEARCH

Loss-of-function of p53 isoform Δ113p53 accelerates brain aging in zebrafish

(2021) Ting Zhao et al.   Cell Death & Disease

Where and Why Modeling Amyotrophic Lateral Sclerosis

(2021) Francesco Liguori et al.   INTERNATIONAL JOURNAL OF MOLECULAR SCIENCES

Molecular Mechanisms Underlying TDP-43 Pathology in Cellular and Animal Models of ALS and FTLD

(2021) Alistair Wood et al.   INTERNATIONAL JOURNAL OF MOLECULAR SCIENCES

C9ORF72: What It Is, What It Does, and Why It Matters

(2021) Julie Smeyers et al.   Frontiers in Cellular Neuroscience

Zebrafish disease models in drug discovery: from preclinical modelling to clinical trials

(2021) E. Elizabeth Patton et al.   NATURE REVIEWS DRUG DISCOVERY

A HML6 endogenous retrovirus on chromosome 3 is upregulated in amyotrophic lateral sclerosis motor cortex

(2021) Ashley R. Jones et al.   Scientific Reports

Reduced C9orf72 function leads to defective synaptic vesicle release and neuromuscular dysfunction in zebrafish

(2021) Zoé Butti et al.   Communications Biology

Nearly 30 Years of Animal Models to Study Amyotrophic Lateral Sclerosis: A Historical Overview and Future Perspectives

(2021) Tiziana Bonifacino et al.   INTERNATIONAL JOURNAL OF MOLECULAR SCIENCES

Recording the ventilation activity of free-swimming zebrafish and its application to novel tank tests

(2021) Masayuki Yoshida   PHYSIOLOGY & BEHAVIOR

Understanding neurobehavioral genetics of zebrafish

(2020) Sergey V. Cheresiz et al.   JOURNAL OF NEUROGENETICS

Functional characterization of a FUS mutant zebrafish line as a novel genetic model for ALS

(2020) Annis-Rayan Bourefis et al.   NEUROBIOLOGY OF DISEASE

IN VITRO AND IN VIVO MODELS OF AMYOTROPHIC LATERAL SCLEROSIS: AN UPDATED OVERVIEW

(2020) Auderlan M. Gois et al.   BRAIN RESEARCH BULLETIN

Lifestyle, Oxidative Stress, and Antioxidants: Back and Forth in the Pathophysiology of Chronic Diseases

(2020) Mehdi Sharifi-Rad et al.   Frontiers in Physiology

Modelling C9orf72-Related Amyotrophic Lateral Sclerosis in Zebrafish

(2020) Gabrielle Fortier et al.   Biomedicines

BMAA and MCLR interact to modulate behavior and exacerbate molecular changes related to neurodegeneration in larval zebrafish

(2020) Rubia M Martin et al.   TOXICOLOGICAL SCIENCES

Developing zebrafish disease models for in vivo small molecule screens

(2019) Pui-Ying Lam et al.   CURRENT OPINION IN CHEMICAL BIOLOGY

ALS and FTD: Where RNA metabolism meets protein quality control

(2019) Jessica Mandrioli et al.   SEMINARS IN CELL & DEVELOPMENTAL BIOLOGY

Lifetime Risk and Heritability of Amyotrophic Lateral Sclerosis

(2019) Marie Ryan et al.   JAMA Neurology

Use of Zebrafish in Drug Discovery Toxicology

(2019) Steven Cassar et al.   CHEMICAL RESEARCH IN TOXICOLOGY

Long‐term edaravone efficacy in amyotrophic lateral sclerosis: Post‐hoc analyses of Study 19 (MCI186‐19)

(2019) Jeremy Shefner et al.   MUSCLE & NERVE

Effect of nutrition on neurodegenerative diseases. A systematic review

(2019) Vittorio Emanuele Bianchi et al.   NUTRITIONAL NEUROSCIENCE

Current basis and future directions of zebrafish nutrigenomics

(2019) Michael B. Williams et al.   Genes and Nutrition

A zebrafish model for C9orf72 ALS reveals RNA toxicity as a pathogenic mechanism

(2018) Bart Swinnen et al.   ACTA NEUROPATHOLOGICA

C9orf72 is essential for neurodevelopment and motility mediated by Cyclin G1

(2018) Tu-Hsueh Yeh et al.   EXPERIMENTAL NEUROLOGY

Expression of C9orf72-related dipeptides impairs motor function in a vertebrate model

(2018) Amrutha Swaminathan et al.   HUMAN MOLECULAR GENETICS

Haploinsufficiency leads to neurodegeneration in C9ORF72 ALS/FTD human induced motor neurons

(2018) Yingxiao Shi et al.   NATURE MEDICINE

Modeling Environmentally-Induced Motor Neuron Degeneration in Zebrafish

(2018) Jessica R. Morrice et al.   Scientific Reports

Excessive daytime sleepiness in Chinese patients with sporadic amyotrophic lateral sclerosis and its association with cognitive and behavioural impairments

(2018) Shuangwu Liu et al.   JOURNAL OF NEUROLOGY NEUROSURGERY AND PSYCHIATRY

Riluzole and edaravone: A tale of two amyotrophic lateral sclerosis drugs

(2018) Manoj Kumar Jaiswal   MEDICINAL RESEARCH REVIEWS

C9orf72-mediated ALS and FTD: multiple pathways to disease

(2018) Rubika Balendra et al.   Nature Reviews Neurology

DT-Diaphorase Prevents Aminochrome-Induced Lysosome Dysfunction in SH-SY5Y Cells

(2018) Catalina Meléndez et al.   NEUROTOXICITY RESEARCH

Inflammation in ALS/FTD pathogenesis

(2018) Madelyn E. McCauley et al.   ACTA NEUROPATHOLOGICA

Mitochondrial superoxide generation induces a parkinsonian phenotype in zebrafish and huntingtin aggregation in human cells

(2018) Brígida R. Pinho et al.   FREE RADICAL BIOLOGY AND MEDICINE

Stable transgenic C9orf72 zebrafish model key aspects of the ALS/FTD phenotype and reveal novel pathological features

(2018) Matthew P. Shaw et al.   Acta Neuropathologica Communications

Induction of oxidative stress by bisphenol A and its pleiotropic effects

(2017) Natalie R. Gassman   ENVIRONMENTAL AND MOLECULAR MUTAGENESIS

Genetic epidemiology of amyotrophic lateral sclerosis: a systematic review and meta-analysis

(2017) Zhang-Yu Zou et al.   JOURNAL OF NEUROLOGY NEUROSURGERY AND PSYCHIATRY

A critical review of the postulated role of the non-essential amino acid, β-N-methylamino-L-alanine, in neurodegenerative disease in humans

(2017) N. Chernoff et al.   JOURNAL OF TOXICOLOGY AND ENVIRONMENTAL HEALTH-PART B-CRITICAL REVIEWS

Safety and efficacy of edaravone in well defined patients with amyotrophic lateral sclerosis: a randomised, double-blind, placebo-controlled trial

(2017) Koji Abe et al.   LANCET NEUROLOGY

Glycine-alanine dipeptide repeat protein contributes to toxicity in a zebrafish model of C9orf72 associated neurodegeneration

(2017) Yu Ohki et al.   Molecular Neurodegeneration

Amyotrophic Lateral Sclerosis

(2017) Robert H. Brown et al.   NEW ENGLAND JOURNAL OF MEDICINE

Little Fish, Big Data: Zebrafish as a Model for Cardiovascular and Metabolic Disease

(2017) Philipp Gut et al.   PHYSIOLOGICAL REVIEWS

Genetics of Amyotrophic Lateral Sclerosis

(2017) Mehdi Ghasemi et al.   Cold Spring Harbor Perspectives in Medicine

Amyotrophic lateral sclerosis

(2017) Orla Hardiman et al.   Nature Reviews Disease Primers

The BSSG rat model of Parkinson’s disease: progressing towards a valid, predictive model of disease

(2017) Jackalina M. Van Kampen et al.   EPMA Journal

Metabolic profiling of zebrafish (Danio rerio) embryos by NMR spectroscopy reveals multifaceted toxicity of β-methylamino-L-alanine (BMAA)

(2017) Upasana Roy et al.   Scientific Reports

p53 isoforms regulate astrocyte-mediated neuroprotection and neurodegeneration

(2016) C Turnquist et al.   CELL DEATH AND DIFFERENTIATION

Sleep Disturbances in Frontotemporal Dementia

(2016) Stuart J. McCarter et al.   Current Neurology and Neuroscience Reports

Drosophilatools and assays for the study of human diseases

(2016) Berrak Ugur et al.   Disease Models & Mechanisms

Military service, deployments, and exposures in relation to amyotrophic lateral sclerosis etiology

(2016) John D. Beard et al.   ENVIRONMENT INTERNATIONAL

Simple animal models for amyotrophic lateral sclerosis drug discovery

(2016) Shunmoogum A. Patten et al.   Expert Opinion on Drug Discovery

Amyotrophic lateral sclerosis: moving towards a new classification system

(2016) Ammar Al-Chalabi et al.   LANCET NEUROLOGY

Chronic Treatment with Paraquat Induces Brain Injury, Changes in Antioxidant Defenses System, and Modulates Behavioral Functions in Zebrafish

(2016) Mauro E. Nunes et al.   MOLECULAR NEUROBIOLOGY

ZNStress: a high-throughput drug screening protocol for identification of compounds modulating neuronal stress in the transgenic mutant sod1G93R zebrafish model of amyotrophic lateral sclerosis

(2016) Alexander McGown et al.   Molecular Neurodegeneration

Loss-of-function genetic tools for animal models: cross-species and cross-platform differences

(2016) Benjamin E. Housden et al.   NATURE REVIEWS GENETICS

Pharmacological modulation of HDAC1 and HDAC6 in vivo in a zebrafish model: Therapeutic implications for Parkinson’s disease

(2016) Brígida R. Pinho et al.   PHARMACOLOGICAL RESEARCH

Dietary exposure to an environmental toxin triggers neurofibrillary tangles and amyloid deposits in the brain

(2016) Paul Alan Cox et al.   PROCEEDINGS OF THE ROYAL SOCIETY B-BIOLOGICAL SCIENCES

Characterization of genetic loss-of-function of Fus in zebrafish

(2016) Svetlana Lebedeva et al.   RNA Biology

Sleep disorders and respiratory function in amyotrophic lateral sclerosis

(2016) Rebekah M. Ahmed et al.   SLEEP MEDICINE REVIEWS

Association of Environmental Toxins With Amyotrophic Lateral Sclerosis

(2016) Feng-Chiao Su et al.   JAMA Neurology

Homology Directed Knockin of Point Mutations in the Zebrafish tardbp and fus Genes in ALS Using the CRISPR/Cas9 System

(2016) Gary Alan Barclay Armstrong et al.   PLoS One

Reverse Genetic Screening Reveals Poor Correlation between Morpholino-Induced and Mutant Phenotypes in Zebrafish

(2015) Fatma O. Kok et al.   DEVELOPMENTAL CELL

Human endogenous retrovirus-K contributes to motor neuron disease

(2015) Wenxue Li et al.   Science Translational Medicine

Out with the old, in with the new: reassessing morpholino knockdowns in light of genome editing technology

(2014) S. Schulte-Merker et al.   DEVELOPMENT

Insights into the Role of the Unusual Disulfide Bond in Copper-Zinc Superoxide Dismutase

(2014) Kevin Sea et al.   JOURNAL OF BIOLOGICAL CHEMISTRY

Spatial and Temporal Control of Transgene Expression in Zebrafish

(2014) Alexander A. Akerberg et al.   PLoS One

Mutant Human FUS Is Ubiquitously Mislocalized and Generates Persistent Stress Granules in Primary Cultured Transgenic Zebrafish Cells

(2014) Jamie Rae Acosta et al.   PLoS One

Zebrafish models of human motor neuron diseases: Advantages and limitations

(2014) Patrick J. Babin et al.   PROGRESS IN NEUROBIOLOGY

Ivermectin: panacea for resource-poor communities?

(2014) Satoshi Ōmura et al.   TRENDS IN PARASITOLOGY

How mitochondrial dysfunction affects zebrafish development and cardiovascular function: anin vivomodel for testing mitochondria-targeted drugs

(2013) Brígida R Pinho et al.   BRITISH JOURNAL OF PHARMACOLOGY

β-N-methylamino-l-alanine causes neurological and pathological phenotypes mimicking Amyotrophic Lateral Sclerosis (ALS): The first step towards an experimental model for sporadic ALS

(2013) Estefanía de Munck et al.   ENVIRONMENTAL TOXICOLOGY AND PHARMACOLOGY

Tardbpl splicing rescues motor neuron and axonal development in a mutant tardbp zebrafish

(2013) Channa A.A. Hewamadduma et al.   HUMAN MOLECULAR GENETICS

The zebrafish reference genome sequence and its relationship to the human genome

(2013) Kerstin Howe et al.   NATURE

Efficient genome editing in zebrafish using a CRISPR-Cas system

(2013) Woong Y Hwang et al.   NATURE BIOTECHNOLOGY

Interaction of FUS and HDAC1 regulates DNA damage response and repair in neurons

(2013) Wen-Yuan Wang et al.   NATURE NEUROSCIENCE

The epidemiology of ALS: a conspiracy of genes, environment and time

(2013) Ammar Al-Chalabi et al.   Nature Reviews Neurology

Pharmacological reduction of ER stress protects against TDP-43 neuronal toxicity in vivo

(2013) Alexandra Vaccaro et al.   NEUROBIOLOGY OF DISEASE

Unconventional Translation of C9ORF72 GGGGCC Expansion Generates Insoluble Polypeptides Specific to c9FTD/ALS

(2013) Peter E.A. Ash et al.   NEURON

Converging Mechanisms in ALS and FTD: Disrupted RNA and Protein Homeostasis

(2013) Shuo-Chien Ling et al.   NEURON

Dietary BMAA Exposure in an Amyotrophic Lateral Sclerosis Cluster from Southern France

(2013) Estelle Masseret et al.   PLoS One

Loss of ALS-associated TDP-43 in zebrafish causes muscle degeneration, vascular dysfunction, and reduced motor neuron axon outgrowth

(2013) B. Schmid et al.   PROCEEDINGS OF THE NATIONAL ACADEMY OF SCIENCES OF THE UNITED STATES OF AMERICA

RAN proteins and RNA foci from antisense transcripts in C9ORF72 ALS and frontotemporal dementia

(2013) T. Zu et al.   PROCEEDINGS OF THE NATIONAL ACADEMY OF SCIENCES OF THE UNITED STATES OF AMERICA

Bisphenol A and human health: A review of the literature

(2013) Johanna R. Rochester   REPRODUCTIVE TOXICOLOGY

The Spinning Task: A New Protocol to Easily Assess Motor Coordination and Resistance in Zebrafish

(2013) Ana R. Blazina et al.   Zebrafish

Towards a Comprehensive Catalog of Zebrafish Behavior 1.0 and Beyond

(2013) Allan V. Kalueff et al.   Zebrafish

Hexanucleotide Repeats in ALS/FTD Form Length-Dependent RNA Foci, Sequester RNA Binding Proteins, and Are Neurotoxic

(2013) Youn-Bok Lee et al.   Cell Reports

Smoking, Alcohol Consumption, and the Risk of Amyotrophic Lateral Sclerosis: A Population-based Study

(2012) S. W. de Jong et al.   AMERICAN JOURNAL OF EPIDEMIOLOGY

Early interneuron dysfunction in ALS: Insights from a mutantsod1zebrafish model

(2012) Alexander McGown et al.   ANNALS OF NEUROLOGY

Time to recognize zebrafish ‘affective’ behavior

(2012) Allan V. Kalueff et al.   BEHAVIOUR

Modeling Neurodegeneration in Zebrafish

(2011) Yanwei Xi et al.   Current Neurology and Neuroscience Reports

Sleep-wake disturbances in patients with amyotrophic lateral sclerosis

(2011) D. Lo Coco et al.   JOURNAL OF NEUROLOGY NEUROSURGERY AND PSYCHIATRY

The syndrome of cognitive impairment in amyotrophic lateral sclerosis: a population-based study

(2011) Julie Phukan et al.   JOURNAL OF NEUROLOGY NEUROSURGERY AND PSYCHIATRY

Heritable gene targeting in zebrafish using customized TALENs

(2011) Peng Huang et al.   NATURE BIOTECHNOLOGY

Targeted gene disruption in somatic zebrafish cells using engineered TALENs

(2011) Jeffry D Sander et al.   NATURE BIOTECHNOLOGY

Clinical diagnosis and management of amyotrophic lateral sclerosis

(2011) Orla Hardiman et al.   Nature Reviews Neurology

Does α-Amino-β-methylaminopropionic Acid (BMAA) Play a Role in Neurodegeneration?

(2011) Alexander S. Chiu et al.   International Journal of Environmental Research and Public Health

Identification of active loci of a human endogenous retrovirus in neurons of patients with amyotrophic lateral sclerosis

(2010) Renée Douville et al.   ANNALS OF NEUROLOGY

A genetic model of amyotrophic lateral sclerosis in zebrafish displays phenotypic hallmarks of motoneuron disease

(2010) T. Ramesh et al.   Disease Models & Mechanisms

Mutant FUS proteins that cause amyotrophic lateral sclerosis incorporate into stress granules

(2010) Daryl A. Bosco et al.   HUMAN MOLECULAR GENETICS

Methylene blue fails to inhibit Tau and polyglutamine protein dependent toxicity in zebrafish

(2010) Frauke van Bebber et al.   NEUROBIOLOGY OF DISEASE

Genetic zebrafish models of neurodegenerative diseases

(2010) Oliver Bandmann et al.   NEUROBIOLOGY OF DISEASE

Progranulin is Neurotrophic In Vivo and Protects against a Mutant TDP-43 Induced Axonopathy

(2010) Angela S. Laird et al.   PLoS One

Identification of a Primary Target of Thalidomide Teratogenicity

(2010) T. Ito et al.   SCIENCE

Toxicity of the cyanobacterial neurotoxin β-N-methylamino-L-alanine to three aquatic animal species

(2009) Esme L. Purdie et al.   Amyotrophic Lateral Sclerosis

Gain and loss of function of ALS-related mutations of TARDBP (TDP-43) cause motor deficits in vivo

(2009) Edor Kabashi et al.   HUMAN MOLECULAR GENETICS

Discovering chemical modifiers of oncogene-regulated hematopoietic differentiation

(2009) Jing-Ruey J Yeh et al.   Nature Chemical Biology

Epidemiology of ALS in Italy: A 10-year prospective population-based study

(2009) A. Chio et al.   NEUROLOGY

Treatment with edaravone, initiated at symptom onset, slows motor decline and decreases SOD1 deposition in ALS mice

(2008) Hidefumi Ito et al.   EXPERIMENTAL NEUROLOGY

Chronic Exposure to Dietary Sterol Glucosides is Neurotoxic to Motor Neurons and Induces an ALS–PDC Phenotype

(2008) R. C. Tabata et al.   NEUROMOLECULAR MEDICINE