Read by QxMD icon Read

autism zebrafish

Federico Sicca, Elena Ambrosini, Maria Marchese, Luigi Sforna, Ilenio Servettini, Giulia Valvo, Maria Stefania Brignone, Angela Lanciotti, Francesca Moro, Alessandro Grottesi, Luigi Catacuzzeno, Sara Baldini, Sonia Hasan, Maria Cristina D'Adamo, Fabio Franciolini, Paola Molinari, Filippo M Santorelli, Mauro Pessia
Dysfunction of the inwardly-rectifying potassium channels Kir4.1 (KCNJ10) represents a pathogenic mechanism contributing to Autism-Epilepsy comorbidity. To define the role of Kir4.1 variants in the disorder, we sequenced KCNJ10 in a sample of affected individuals, and performed genotype-phenotype correlations. The effects of mutations on channel activity, protein trafficking, and astrocyte function were investigated in Xenopus laevis oocytes, and in human astrocytoma cell lines. An in vivo model of the disorder was also explored through generation of kcnj10a morphant zebrafish overexpressing the mutated human KCNJ10...
September 28, 2016: Scientific Reports
Yuanchuan Wang, Hongxiu Zhong, Chonggang Wang, Dongxu Gao, Yulin Zhou, Zhenghong Zuo
Autism spectrum disorder (ASD) is a serious debilitating mental illness with complex symptoms and multi-factorial pathogenesis. Although the pathogenesis of ASD remains unclear, etiology is thought to involve complex, multigenic interactions and possible environmental contributions. In the present study, we used zebrafish (Danio rerio) as a model to investigate whether maternal exposure to the water soluble fraction of crude oil (WSF, 5μg/L), lead (Pb, 20μg/L) and their mixture (5 μg/L WSF+20 μg/L Pb) could induce autism-like behavior in larvae...
August 26, 2016: Ecotoxicology and Environmental Safety
Chun-Xue Liu, Xiao-Lan Peng, Chun-Chun Hu, Chun-Yang Li, Qiang Li, Xiu Xu
SHANK3 is a scaffolding protein that binds to various synaptic proteins at the postsynaptic density (PSD) of excitatory glutamatergic synapses. SHANK3 is not only strongly implicated in autism spectrum disorders (ASD) but also plays a critical role in human Phelan-McDermid syndrome (22q13.3 deletion syndrome). Accumulated experimental evidence demonstrates that the zebrafish model system is useful for studying the functions of ASD-related gene during early development. However, many basic features of shank3 transcript expression in zebrafish remain poorly understood...
August 26, 2016: Development Genes and Evolution
Robert A Kozol, Alexander J Abrams, David M James, Elena Buglo, Qing Yan, Julia E Dallman
Zebrafish are a unique cell to behavior model for studying the basic biology of human inherited neurological conditions. Conserved vertebrate genetics and optical transparency provide in vivo access to the developing nervous system as well as high-throughput approaches for drug screens. Here we review zebrafish modeling for two broad groups of inherited conditions that each share genetic and molecular pathways and overlap phenotypically: neurodevelopmental disorders such as Autism Spectrum Disorders (ASD), Intellectual Disability (ID) and Schizophrenia (SCZ), and neurodegenerative diseases, such as Cerebellar Ataxia (CATX), Hereditary Spastic Paraplegia (HSP) and Charcot-Marie Tooth Disease (CMT)...
2016: Frontiers in Molecular Neuroscience
Fernanda Francine Zimmermann, Karina Vidarte Gaspary, Anna Maria Siebel, Carla Denise Bonan
Changes in social behavior occur in several neuropsychiatric disorders such as schizophrenia and autism. The interaction between individuals is an essential aspect and an adaptive response of several species, among them the zebrafish. Oxytocin is a neuroendocrine hormone associated with social behavior. The aim of the present study was to investigate the effects of MK-801, a non-competitive antagonist of glutamate NMDA receptors, on social interaction and aggression in zebrafish. We also examined the modulation of those effects by oxytocin, the oxytocin receptor agonist carbetocin and the oxytocin receptor antagonist L-368,899...
September 15, 2016: Behavioural Brain Research
Fernanda Francine Zimmermann, Karina Vidarte Gaspary, Anna Maria Siebel, Carlos Eduardo Leite, Luiza Wilges Kist, Mauricio Reis Bogo, Carla Denise Bonan
Autism is a neurodevelopmental disorder characterized by symptoms related to stereotyped movements, deficits in social interaction, impaired communication, anxiety, hyperactivity, and the presence of restricted interests. Evidence indicates an important role of extracellular ATP and adenosine as signaling molecules in autism. ATP hydrolysis by ectonucleotidases is an important source of adenosine, and adenosine deaminase (ADA) contributes to the control of the nucleoside concentrations. Considering zebrafish is an animal model that may contribute towards to understanding the mechanisms that underlie social behavior, we investigated the purinergic signaling in a model of embryological exposure to valproic acid (VPA) that induces social interaction deficit in adult zebrafish...
May 17, 2016: Molecular Neurobiology
Jonathan D J Labonne, Kang-Han Lee, Shigeki Iwase, Il-Keun Kong, Michael P Diamond, Lawrence C Layman, Cheol-Hee Kim, Hyung-Goo Kim
Microdeletion syndromes are frequent causes of neuropsychiatric disorders leading to intellectual disability as well as autistic features accompanied by epilepsy and craniofacial anomalies. From comparative deletion mapping of the smallest microdeletion to date at 12q24.31, found in a patient with overlapping clinical features of 12q24.31 microdeletion syndrome, we narrowed the putative critical region to 445 kb containing seven genes, one microRNA, and one non-coding RNA. Zebrafish in situ hybridization and comprehensive transcript analysis of annotated genes in the panels of human organ and brain suggest that these are all candidates for neurological phenotypes excluding the gene HPD...
July 2016: Human Genetics
Brieana Fregeau, Bum Jun Kim, Andrés Hernández-García, Valerie K Jordan, Megan T Cho, Rhonda E Schnur, Kristin G Monaghan, Jane Juusola, Jill A Rosenfeld, Elizabeth Bhoj, Elaine H Zackai, Stephanie Sacharow, Kristin Barañano, Daniëlle G M Bosch, Bert B A de Vries, Kristin Lindstrom, Audrey Schroeder, Philip James, Peggy Kulch, Seema R Lalani, Mieke M van Haelst, Koen L I van Gassen, Ellen van Binsbergen, A James Barkovich, Daryl A Scott, Elliott H Sherr
Deletions of chromosome 1p36 affect approximately 1 in 5,000 newborns and are associated with developmental delay, intellectual disability, and defects involving the brain, eye, ear, heart, and kidney. Arginine-glutamic acid dipeptide repeats (RERE) is located in the proximal 1p36 critical region. RERE is a widely-expressed nuclear receptor coregulator that positively regulates retinoic acid signaling. Animal models suggest that RERE deficiency might contribute to many of the structural and developmental birth defects and medical problems seen in individuals with 1p36 deletion syndrome, although human evidence supporting this role has been lacking...
May 5, 2016: American Journal of Human Genetics
Elise Heon, Gunhee Kim, Sophie Qin, Janelle E Garrison, Erika Tavares, Ajoy Vincent, Nina Nuangchamnong, C Anthony Scott, Diane C Slusarski, Val C Sheffield
Bardet Biedl syndrome (BBS) is a multisystem genetically heterogeneous ciliopathy that most commonly leads to obesity, photoreceptor degeneration, digit anomalies, genito-urinary abnormalities, as well as cognitive impairment with autism, among other features. Sequencing of a DNA sample from a 17 year old female affected with BBS did not identify any mutation in the known BBS genes. Whole genome sequencing identified a novel loss-of-function disease-causing homozygous mutation (K102*) inC8ORF37, a gene coding for a cilia protein...
March 22, 2016: Human Molecular Genetics
Jakob Biran, Gil Levkowitz
Chemical genetics can help decipher novel pathways underlying neurodevelopmental psychiatric impairments. Hoffman et al. (2016) utilized behavioral profiling of psychoactive compounds in zebrafish and identified estrogens as suppressors of a phenotype resulting from loss of an autism risk gene.
February 17, 2016: Neuron
Simon Perathoner, Maria Lorena Cordero-Maldonado, Alexander D Crawford
Emotion is a key aspect of behavior, enabling humans and animals to assign either positive or negative values to sensory inputs and thereby to make appropriate decisions. Classical experiments in mammalian models, mainly in primates and rodents, have shown that the amygdala is essential for appetitive and aversive associative processing and that dysfunction of this brain region leads to various psychiatric conditions, including depression, generalized anxiety disorder, panic disorder, phobias, autism, and posttraumatic stress disorder...
June 2016: Journal of Neuroscience Research
Ellen J Hoffman, Katherine J Turner, Joseph M Fernandez, Daniel Cifuentes, Marcus Ghosh, Sundas Ijaz, Roshan A Jain, Fumi Kubo, Brent R Bill, Herwig Baier, Michael Granato, Michael J F Barresi, Stephen W Wilson, Jason Rihel, Matthew W State, Antonio J Giraldez
Autism spectrum disorders (ASDs) are a group of devastating neurodevelopmental syndromes that affect up to 1 in 68 children. Despite advances in the identification of ASD risk genes, the mechanisms underlying ASDs remain unknown. Homozygous loss-of-function mutations in Contactin Associated Protein-like 2 (CNTNAP2) are strongly linked to ASDs. Here we investigate the function of Cntnap2 and undertake pharmacological screens to identify phenotypic suppressors. We find that zebrafish cntnap2 mutants display GABAergic deficits, particularly in the forebrain, and sensitivity to drug-induced seizures...
February 17, 2016: Neuron
Shota Sasagawa, Yuhei Nishimura, Junko Koiwa, Tsuyoshi Nomoto, Taichi Shintou, Soichiro Murakami, Mizuki Yuge, Koki Kawaguchi, Reiko Kawase, Takeshi Miyazaki, Toshio Tanaka
Mitochondrial dysfunction has been implicated in various drug-induced toxicities and genetic disorders. Recently, the zebrafish has emerged as a versatile animal model for both chemical and genetic screenings. Taking advantage of its transparency, various in vivo fluorescent imaging methods have been developed to identify novel functions of chemicals and genes in zebrafish. However, there have not been fluorescent probes that can detect mitochondrial membrane potential in living zebrafish. In this study, we identified a novel cyanine dye called ZMJ214 that detects mitochondrial membrane potential in living zebrafish from 4 to 8 days post fertilization and is administered by simple immersion...
February 19, 2016: ACS Chemical Biology
Xuedong Wang, Yuansi Zheng, Yuna Zhang, Jieyi Li, Hongqin Zhang, Huili Wang
To date, few data are available on neurotoxicity of β-diketone antibiotics (DKAs) from the perspective of animal behavior. Herein, the effects of long-term DKAs exposure on zebrafish (Danio rerio) behavior were assessed for locomotor activity, anxiety, social interaction and their related molecular mechanisms. DKAs exposure to zebrafish consisted of six DKA species, including ofloxacin, ciprofloxacin, enrofloxacin, doxycycline, chlortetracycline and oxytetracycline, with equal weight concentration and equal volume...
February 2016: Chemosphere
Ava K Lovato, Robbert Creton, Ruth M Colwill
Developmental disorders such as anxiety, autism, and attention deficit hyperactivity disorders have been linked to exposure to polychlorinated biphenyls (PCBs), a ubiquitous anthropogenic pollutant. The zebrafish is widely recognized as an excellent model system for assessing the effects of toxicant exposure on behavior and neurodevelopment. In the present study, we examined the effect of sub-chronic embryonic exposure to the PCB mixture, Aroclor (A) 1254 on anxiety-related behaviors in zebrafish larvae at 7 days post-fertilization (dpf)...
January 2016: Neurotoxicology and Teratology
Lingyan Xing, Jong-Hyun Son, Tamara J Stevenson, Christina Lillesaar, Laure Bally-Cuif, Tiffanie Dahl, Joshua L Bonkowsky
UNLABELLED: Modulation of connectivity formation in the developing brain in response to external stimuli is poorly understood. Here, we show that the raphe nucleus and its serotonergic projections regulate pathfinding of commissural axons in zebrafish. We found that the raphe neurons extend projections toward midline-crossing axons and that when serotonergic signaling is blocked by pharmacological inhibition or by raphe neuron ablation, commissural pathfinding is disrupted. We demonstrate that the serotonin receptor htr2a is expressed on these commissural axons and that genetic knock-down of htr2a disrupts crossing...
November 4, 2015: Journal of Neuroscience: the Official Journal of the Society for Neuroscience
Fernanda Francine Zimmermann, Karina Vidarte Gaspary, Carlos Eduardo Leite, Giana De Paula Cognato, Carla Denise Bonan
Changes in social behavior are associated with brain disorders, including mood disorders, stress, schizophrenia, Alzheimer's disease, and autism spectrum disorders (ASD). Autism is a complex neurodevelopmental disorder characterized by deficits in social interaction, impaired communication, anxiety, hyperactivity, and the presence of restricted interests. Zebrafish is one of the most social vertebrates used as a model in biomedical research, contributing to an understanding of the mechanisms that underlie social behavior...
November 2015: Neurotoxicology and Teratology
M Pilorge, C Fassier, H Le Corronc, A Potey, J Bai, S De Gois, E Delaby, B Assouline, V Guinchat, F Devillard, R Delorme, G Nygren, M Råstam, J C Meier, S Otani, H Cheval, V M James, M Topf, T N Dear, C Gillberg, M Leboyer, B Giros, S Gautron, J Hazan, R J Harvey, P Legendre, C Betancur
Autism spectrum disorder (ASD) is a common neurodevelopmental condition characterized by marked genetic heterogeneity. Recent studies of rare structural and sequence variants have identified hundreds of loci involved in ASD, but our knowledge of the overall genetic architecture and the underlying pathophysiological mechanisms remains incomplete. Glycine receptors (GlyRs) are ligand-gated chloride channels that mediate inhibitory neurotransmission in the adult nervous system but exert an excitatory action in immature neurons...
July 2016: Molecular Psychiatry
Daniela Edith Igartúa, María Natalia Calienni, Daniela Agustina Feas, Nadia Silvia Chiaramoni, Silvia Del Valle Alonso, María Jimena Prieto
Emulsions are gaining increasing interest to be applied as drug delivery systems. The main goal of this work was the formulation of an oil/water nutraceutical emulsion (NE) for oral administration, enriched in omega 3 (ω3) and omega 6 (ω6), and able to encapsulate risperidone (RISP), an antipsychotic drug widely used in the treatment of autism spectrum disorders (ASD). RISP has low solubility in aqueous medium and poor bioavailability because of its metabolism and high protein binding. Coadministration of ω3, ω3, and vitamin E complexed with RISP might increase its bioavailability and induce a synergistic effect on the treatment of ASD...
December 2015: Journal of Pharmaceutical Sciences
Juan Du, Wei Lü, Shenping Wu, Yifan Cheng, Eric Gouaux
The strychnine-sensitive glycine receptor (GlyR) mediates inhibitory synaptic transmission in the spinal cord and brainstem and is linked to neurological disorders, including autism and hyperekplexia. Understanding of molecular mechanisms and pharmacology of glycine receptors has been hindered by a lack of high-resolution structures. Here we report electron cryo-microscopy structures of the zebrafish α1 GlyR with strychnine, glycine, or glycine and ivermectin (glycine/ivermectin). Strychnine arrests the receptor in an antagonist-bound closed ion channel state, glycine stabilizes the receptor in an agonist-bound open channel state, and the glycine/ivermectin complex adopts a potentially desensitized or partially open state...
October 8, 2015: Nature
Fetch more papers »
Fetching more papers... Fetching...
Read by QxMD. Sign in or create an account to discover new knowledge that matter to you.
Remove bar
Read by QxMD icon Read

Search Tips

Use Boolean operators: AND/OR

diabetic AND foot
diabetes OR diabetic

Exclude a word using the 'minus' sign

Virchow -triad

Use Parentheses

water AND (cup OR glass)

Add an asterisk (*) at end of a word to include word stems

Neuro* will search for Neurology, Neuroscientist, Neurological, and so on

Use quotes to search for an exact phrase

"primary prevention of cancer"
(heart or cardiac or cardio*) AND arrest -"American Heart Association"