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Zebrafish disease modelling

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https://www.readbyqxmd.com/read/28917662/successful-optic-nerve-regeneration-in-the-senescent-zebrafish-despite-age-related-decline-of-cell-intrinsic-and-extrinsic-response-processes
#1
Jessie Van Houcke, Ilse Bollaerts, Emiel Geeraerts, Benjamin Davis, An Beckers, Inge Van Hove, Kim Lemmens, Lies De Groef, Lieve Moons
Dysfunction of the central nervous system (CNS) in neurodegenerative diseases or after brain lesions seriously affects life quality of a growing number of elderly, since the adult CNS lacks the capacity to replace or repair damaged neurons. Despite intensive research efforts, full functional recovery after CNS disease and/or injury remains challenging, especially in an aging environment. As such, there is a rising need for an aging model in which the impact of aging on successful regeneration can be studied...
August 24, 2017: Neurobiology of Aging
https://www.readbyqxmd.com/read/28914763/emerging-estrogenic-pollutants-in-the-aquatic-environment-and-breast-cancer
#2
REVIEW
Sylvain Lecomte, Denis Habauzit, Thierry D Charlier, Farzad Pakdel
The number and amount of man-made chemicals present in the aquatic environment has increased considerably over the past 50 years. Among these contaminants, endocrine-disrupting chemicals (EDCs) represent a significant proportion. This family of compounds interferes with normal hormonal processes through multiple molecular pathways. They represent a potential risk for human and wildlife as they are suspected to be involved in the development of diseases including, but not limited to, reprotoxicity, metabolic disorders, and cancers...
September 15, 2017: Genes
https://www.readbyqxmd.com/read/28912673/improving-the-delivery-of-sod1-antisense-oligonucleotides-to-motor-neurons-using-calcium-phosphate-lipid-nanoparticles
#3
Liyu Chen, Clare Watson, Marco Morsch, Nicholas J Cole, Roger S Chung, Darren N Saunders, Justin J Yerbury, Kara L Vine
Amyotrophic Lateral Sclerosis (ALS) is a fatal neurodegenerative disease affecting the upper and lower motor neurons in the motor cortex and spinal cord. Abnormal accumulation of mutant superoxide dismutase I (SOD1) in motor neurons is a pathological hallmark of some forms of the disease. We have shown that the orderly progression of the disease may be explained by misfolded SOD1 cell-to-cell propagation, which is reliant upon its active endogenous synthesis. Reducing the levels of SOD1 is therefore a promising therapeutic approach...
2017: Frontiers in Neuroscience
https://www.readbyqxmd.com/read/28911205/a-zebrafish-model-of-x-linked-adrenoleukodystrophy-recapitulates-key-disease-features-and-demonstrates-a-developmental-requirement-for-abcd1-in-oligodendrocyte-patterning-and-myelination
#4
Lauren R Strachan, Tamara J Stevenson, Briana Freshner, Matthew D Keefe, D Miranda Bowles, Joshua L Bonkowsky
X-linked adrenoleukodystrophy (ALD) is a devastating inherited neurodegenerative disease caused by defects in the ABCD1 gene and affecting peripheral and central nervous system myelin. ABCD1 encodes a peroxisomal transmembrane protein required for very long chain fatty acid (VLCFA) metabolism. We show that zebrafish (Danio rerio) Abcd1 is highly conserved at the amino acid level with human ABCD1, and during development is expressed in homologous regions including the central nervous system and adrenal glands...
September 15, 2017: Human Molecular Genetics
https://www.readbyqxmd.com/read/28910236/quantitative-responses-of-adult-zebrafish-to-changes-in-ambient-illumination
#5
Enhua Shao, Qing Bai, Yangzhong Zhou, Edward A Burton
The use of zebrafish models to study central nervous system aging and late-onset neurological diseases will be facilitated by assays allowing rapid evaluation of neurological phenotypes in adult zebrafish. We analyzed groups of 12 adult zebrafish swimming simultaneously in single-animal arenas, and quantified their responses to changes in ambient illumination. Under these conditions, stereotypical locomotor patterns were observed and readily quantified using open source software. Continuous, low-velocity movements were observed during 10-min periods of darkness, whereas intermittent high-velocity movements occurred in bright light...
September 14, 2017: Zebrafish
https://www.readbyqxmd.com/read/28901286/biochemical-adaptations-of-the-retina-and-retinal-pigment-epithelium-support-a-metabolic-ecosystem-in-the-vertebrate-eye
#6
Mark A Kanow, Michelle M Giarmarco, Connor Sr Jankowski, Kristine Tsantilas, Abbi L Engel, Jianhai Du, Jonathan D Linton, Christopher C Farnsworth, Stephanie R Sloat, Austin Rountree, Ian R Sweet, Ken J Lindsay, Edward D Parker, Susan E Brockerhoff, Martin Sadilek, Jennifer R Chao, James B Hurley
Here we report multiple lines of evidence for a comprehensive model of energy metabolism in the vertebrate eye. Metabolic flux, locations of key enzymes, and our finding that glucose enters mouse and zebrafish retinas mostly through photoreceptors support a conceptually new model for retinal metabolism. In this model, glucose from the choroidal blood passes through the retinal pigment epithelium to the retina where photoreceptors convert it to lactate. Photoreceptors then export the lactate as fuel for the retinal pigment epithelium and for neighboring Müller glial cells...
September 13, 2017: ELife
https://www.readbyqxmd.com/read/28899790/alisporivir-rescues-defective-mitochondrial-respiration-in-duchenne-muscular-dystrophy
#7
Marco Schiavone, Alessandra Zulian, Sara Menazza, Valeria Petronilli, Francesco Argenton, Luciano Merlini, Patrizia Sabatelli, Paolo Bernardi
Duchenne muscular dystrophy (DMD) is a severe muscle disease of known etiology without effective, or generally applicable therapy. Mitochondria are affected by the disease in animal models but whether mitochondrial dysfunction is part of the pathogenesis in patients remains unclear. We show that primary cultures obtained from muscle biopsies of DMD patients display a decrease of the respiratory reserve, a consequence of inappropriate opening of the permeability transition pore (PTP). Treatment with the cyclophilin inhibitor alisporivir - a cyclosporin A derivative that desensitizes the PTP but does not inhibit calcineurin - largely restored the maximal respiratory capacity without affecting basal oxygen consumption in cells from patients, thus reinstating a normal respiratory reserve...
September 9, 2017: Pharmacological Research: the Official Journal of the Italian Pharmacological Society
https://www.readbyqxmd.com/read/28898690/when-lmo1-meets-mycn-neuroblastoma-is-metastatic
#8
Zhihui Liu, Carol J Thiele
LMO1 is a high-risk neuroblastoma susceptibility gene, but how LMO1 cooperates with MYCN in neuroblastoma tumorigenesis is unclear. In this issue of Cancer Cell, Zhu et al. develop a novel zebrafish model that elucidates a mechanism by which LMO1 and MYCN synergistically initiate neuroblastoma and contribute to metastatic disease progression.
September 11, 2017: Cancer Cell
https://www.readbyqxmd.com/read/28890853/preferences-for-achromatic-horizontal-vertical-and-square-patterns-in-zebrafish-danio-rerio
#9
Lisa A Rimstad, Adam Holcombe, Alicia Pope, Trevor J Hamilton, Melike P Schalomon
The zebrafish (Danio rerio) is gaining popularity as a laboratory organism and is used to model many human diseases. Many behavioural measures of locomotion and cognition have been developed that involve the processing of visual stimuli. However, the innate preference for vertical and horizontal stripes in zebrafish is unknown. We tested the preference of adult zebrafish for three achromatic patterns (vertical stripes, horizontal stripes, and squares) at three different size conditions (1, 5, and 10 mm). Each animal was tested once in a rectangular arena, which had a different pattern of the same size condition on the walls of either half of the arena...
2017: PeerJ
https://www.readbyqxmd.com/read/28888969/mtor-related-neuropathology-in-mutant-tsc2-zebrafish-phenotypic-transcriptomic-and-pharmacological-analysis
#10
Chloë Scheldeman, James D Mills, Aleksandra Siekierska, Ines Serra, Daniëlle Copmans, Anand M Iyer, Benjamin J Whalley, Jan Maes, Anna C Jansen, Lieven Lagae, Eleonora Aronica, Peter A M de Witte
Tuberous sclerosis complex (TSC) is a rare, genetic disease caused by loss-of-function mutations in either TSC1 or TSC2. Patients with TSC are neurologically characterized by the presence of abnormal brain structure, intractable epilepsy and TSC-associated neuropsychiatric disorders. Given the lack of effective long-term treatments for TSC, there is a need to gain greater insight into TSC-related pathophysiology and to identify and develop new treatments. In this work we show that homozygous tsc2(-/-) mutant zebrafish larvae, but not tsc2(+/-) and WT larvae, display enlarged brains, reduced locomotor behavior and epileptiform discharges at 7dpf...
September 6, 2017: Neurobiology of Disease
https://www.readbyqxmd.com/read/28888214/catching-new-targets-in-metabolic-disease-with-a-zebrafish
#11
REVIEW
Margrit Kamel, Nikolay Ninov
Traditionally, the development of novel therapeutics for metabolic diseases has relied mainly on high-throughput screening using biochemical or cell-based assays. While this approach represents a driving force in drug discovery, there is also a need to perform large-scale screens without disrupting inter-organ communication and tissue architecture, essential components for understanding the complexity of metabolic regulation and the identification of small molecules with appropriate biological activities in vivo...
September 6, 2017: Current Opinion in Pharmacology
https://www.readbyqxmd.com/read/28884682/microct-based-phenomics-in-the-zebrafish-skeleton-reveals-virtues-of-deep-phenotyping-in-a-distributed-organ-system
#12
Matthew Hur, Charlotte A Gistelinck, Philippe Huber, Jane Lee, Marjorie H Thompson, Adrian T Monstad-Rios, Claire J Watson, Sarah K McMenamin, Andy Willaert, David M Parichy, Paul Coucke, Ronald Y Kwon
Phenomics, which ideally involves in-depth phenotyping at the whole-organism scale, may enhance our functional understanding of genetic variation. Here, we demonstrate methods to profile hundreds of phenotypic measures comprised of morphological and densitometric traits at a large number of sites within the axial skeleton of adult zebrafish. We show the potential for vertebral patterns to confer heightened sensitivity, with similar specificity, in discriminating mutant populations compared to analyzing individual vertebrae in isolation...
September 8, 2017: ELife
https://www.readbyqxmd.com/read/28882225/animal-models-to-study-microrna-function
#13
Arpita S Pal, Andrea L Kasinski
The discovery of the microRNAs, lin-4 and let-7 as critical mediators of normal development in Caenorhabditis elegans and their conservation throughout evolution has spearheaded research toward identifying novel roles of microRNAs in other cellular processes. To accurately elucidate these fundamental functions, especially in the context of an intact organism, various microRNA transgenic models have been generated and evaluated. Transgenic C. elegans (worms), Drosophila melanogaster (flies), Danio rerio (zebrafish), and Mus musculus (mouse) have contributed immensely toward uncovering the roles of multiple microRNAs in cellular processes such as proliferation, differentiation, and apoptosis, pathways that are severely altered in human diseases such as cancer...
2017: Advances in Cancer Research
https://www.readbyqxmd.com/read/28882119/aggregation-prone-gfap-mutation-in-alexander-disease-validated-using-a-zebrafish-model
#14
So-Hyun Lee, Tai-Seung Nam, Kun-Hee Kim, Jin Hee Kim, Woong Yoon, Suk-Hee Heo, Min Jung Kim, Boo Ahn Shin, Ming-Der Perng, Hyon E Choy, Jihoon Jo, Myeong-Kyu Kim, Seok-Yong Choi
BACKGROUND: Alexander disease (AxD) is an astrogliopathy that predominantly affects the white matter of the central nervous system (CNS), and is caused by a mutation in the gene encoding the glial fibrillary acidic protein (GFAP), an intermediate filament primarily expressed in astrocytes and ependymal cells. The main pathologic feature of AxD is the presence of Rosenthal fibers (RFs), homogeneous eosinophilic inclusions found in astrocytes. Because of difficulties in procuring patient' CNS tissues and the presence of RFs in other pathologic conditions, there is a need to develop an in vivo assay that can determine whether a mutation in the GFAP results in aggregation and is thus disease-causing...
September 7, 2017: BMC Neurology
https://www.readbyqxmd.com/read/28880915/an-optimized-method-for-counting-dopaminergic-neurons-in-zebrafish
#15
Hideaki Matsui, Atsushi Sugie
In recent years, considerable effort has been devoted to the development of a fish model for Parkinson's disease (PD) to examine the pathological mechanisms of neurodegeneration. To effectively evaluate PD pathology, the ability to accurately and reliably count dopaminergic neurons is important. However, there is currently no such standardized method. Due to the relatively small number of dopaminergic neurons in fish, stereological estimation would not be suitable. In addition, serial sectioning requires proficiency to not lose any sections, and it permits double counting due to the large size of some of the dopaminergic neurons...
2017: PloS One
https://www.readbyqxmd.com/read/28880735/2017-midwest-zebrafish-meeting-report
#16
Elizabeth Sandquist, Sarah C Petersen, Cody J Smith
The 2017 Midwest Zebrafish meeting was held from June 16 to 18 at the University of Cincinnati, sponsored by the Cincinnati Children's Hospital Divisions of Developmental Biology, Molecular Cardiovascular Biology, and Gastroenterology, Hepatology, and Nutrition. The meeting, organized by Saulius Sumanas, Joshua Waxman, and Chunyue Yin, hosted >130 attendees from 16 different states. Scientific sessions were focused on morphogenesis, neural development, novel technologies, and disease models, with Steve Ekker, Stephen Potter, and Lila Solnica-Krezel presenting keynote talks...
September 7, 2017: Zebrafish
https://www.readbyqxmd.com/read/28874875/the-role-of-histone-modification-and-a-regulatory-single-nucleotide-polymorphism-rs2071166-in-the-cx43-promoter-in-patients-with-tof
#17
Ruoyi Gu, Jun Xu, Yixiang Lin, Wei Sheng, Duan Ma, Xiaojing Ma, Guoying Huang
Abnormal level of Cx43 expression could result in CHD. Epigenetic modification and disease-associated, non-coding SNPs might influence gene transcription and expression. Our study aimed to determine the role of histone modification and an rSNP (rs2071166) in the Cx43 promoter in patients with TOF. Our results indicate that H3K18ac bind to Cx43 promoter and that their levels are reduced in TOF patients relative to controls. The relationship between the non-coding SNP in the Cx43 gene and TOF patients was evaluated in 158 patients and 300 controls...
September 5, 2017: Scientific Reports
https://www.readbyqxmd.com/read/28873048/gene-ontology-based-analysis-of-zebrafish-omics-data-using-the-web-tool-comparative-gene-ontology
#18
Esmaeil Ebrahimie, Mario Fruzangohar, Seyyed Hani Moussavi Nik, Morgan Newman
Gene Ontology (GO) analysis is a powerful tool in systems biology, which uses a defined nomenclature to annotate genes/proteins within three categories: "Molecular Function," "Biological Process," and "Cellular Component." GO analysis can assist in revealing functional mechanisms underlying observed patterns in transcriptomic, genomic, and proteomic data. The already extensive and increasing use of zebrafish for modeling genetic and other diseases highlights the need to develop a GO analytical tool for this organism...
September 5, 2017: Zebrafish
https://www.readbyqxmd.com/read/28872594/zebrafish-xenograft-an-evolutionary-experiment-in-tumour-biology
#19
REVIEW
Rachael A Wyatt, Nhu P V Trieu, Bryan D Crawford
Though the cancer research community has used mouse xenografts for decades more than zebrafish xenografts, zebrafish have much to offer: they are cheap, easy to work with, and the embryonic model is relatively easy to use in high-throughput assays. Zebrafish can be imaged live, allowing us to observe cellular and molecular processes in vivo in real time. Opponents dismiss the zebrafish model due to the evolutionary distance between zebrafish and humans, as compared to mice, but proponents argue for the zebrafish xenograft's superiority to cell culture systems and its advantages in imaging...
September 5, 2017: Genes
https://www.readbyqxmd.com/read/28867638/zebrafish-a-promising-in-vivo-model-for-assessing-the-delivery-of-natural-products-fluorescence-dyes-and-drugs-across-the-blood-brain-barrier
#20
REVIEW
Ye Li, Tongkai Chen, Xiaoqing Miao, Xiang Yi, Xueqing Wang, Haitao Zhao, Simon Ming-Yuen Lee, Ying Zheng
The blood brain barrier (BBB) is the network of capillaries that controls the passage of substances from the blood into the brain and other parts of the central nervous system (CNS). As this barrier is the major obstacle for drug delivery into CNS, a credible BBB model is very necessary to assess the BBB permeability of novel neuroactive compounds including thousands of bioactive compounds which have been extracted from medicinal plants and have the potential for the treatment of CNS diseases. Increasing reports indicated that zebrafish has emerged as a timely, reproducible model for BBB permeability assessment...
August 31, 2017: Pharmacological Research: the Official Journal of the Italian Pharmacological Society
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