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Zebrafish disease modelling

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https://www.readbyqxmd.com/read/29772372/palmitate-induces-nitric-oxide-production-and-inflammatory-cytokine-expression-in-zebrafish
#1
Seon-Heui Cha, Yongha Hwang, Kil-Nam Kim, Hee-Sook Jun
Inflammation markers in zebrafish embryos reflect a toxic response that is common to other animal models and humans. Free fatty acids (FFAs) are known to cause damage in various tissues by inducing inflammation. In this study, we investigated whether a FFA (palmitate) induces inflammation in zebrafish embryos. Nitrous oxide (NO) production and cyclooxygenase-2 (COX-2) mRNA expression were increased in palmitate-treated zebrafish embryos in a dose-dependent manner. mRNA expression of pro-inflammatory cytokines, interleukin-1β (IL-1β) and tumor necrosis factor-α (TNF- α), were also increased...
May 14, 2018: Fish & Shellfish Immunology
https://www.readbyqxmd.com/read/29764394/comprehensive-analysis-of-coding-lncrna-gene-co-expression-network-uncovers-conserved-functional-lncrnas-in-zebrafish
#2
Wen Chen, Xuan Zhang, Jing Li, Shulan Huang, Shuanglin Xiang, Xiang Hu, Changning Liu
BACKGROUND: Zebrafish is a full-developed model system for studying development processes and human disease. Recent studies of deep sequencing had discovered a large number of long non-coding RNAs (lncRNAs) in zebrafish. However, only few of them had been functionally characterized. Therefore, how to take advantage of the mature zebrafish system to deeply investigate the lncRNAs' function and conservation is really intriguing. RESULTS: We systematically collected and analyzed a series of zebrafish RNA-seq data, then combined them with resources from known database and literatures...
May 9, 2018: BMC Genomics
https://www.readbyqxmd.com/read/29761463/using-zfin-data-types-organization-and-retrieval
#3
Ceri E Van Slyke, Yvonne M Bradford, Douglas G Howe, David S Fashena, Sridhar Ramachandran, Leyla Ruzicka
The Zebrafish Model Organism Database (ZFIN; zfin.org) was established in 1994 as the primary genetic and genomic resource for the zebrafish research community. Some of the earliest records in ZFIN were for people and laboratories. Since that time, services and data types provided by ZFIN have grown considerably. Today, ZFIN provides the official nomenclature for zebrafish genes, mutants, and transgenics and curates many data types including gene expression, phenotypes, Gene Ontology, models of human disease, orthology, knockdown reagents, transgenic constructs, and antibodies...
2018: Methods in Molecular Biology
https://www.readbyqxmd.com/read/29757277/profiling-dna-replication-timing-using-zebrafish-as-an-in-vivo-model-system
#4
Joseph C Siefert, Emily A Clowdus, Duane Goins, Amnon Koren, Christopher L Sansam
DNA replication timing is an important cellular characteristic, exhibiting significant relationships with chromatin structure, transcription, and DNA mutation rates. Changes in replication timing occur during development and in cancer, but the role replication timing plays in development and disease is not known. Zebrafish were recently established as an in vivo model system to study replication timing. Here is detailed the protocols for using the zebrafish to determine DNA replication timing. After sorting cells from embryos and adult zebrafish, high-resolution genome-wide DNA replication timing patterns can be constructed by determining changes in DNA copy number through analysis of next generation sequencing data...
April 30, 2018: Journal of Visualized Experiments: JoVE
https://www.readbyqxmd.com/read/29753045/identification-and-functional-characterization-of-zebrafish-k-2p-17-1-task-4-talk-2-two-pore-domain-k-channels
#5
Ingo Staudacher, Claudius Illg, Jakob Gierten, Sebastian Seehausen, Patrick A Schweizer, Hugo A Katus, Dierk Thomas
Human K2P 17.1 (TASK-4, TALK-2) two-pore-domain potassium (K2P ) channels have recently been implicated in heart rhythm disorders including atrial fibrillation and conduction disease. The functional in vivo significance of K2P 17.1 currents in cardiac electrophysiology remains incompletely understood. Danio rerio (zebrafish) may be utilized to elucidate the role of cardiac K2P channels in vivo. The aim of this work was to identify and characterize the zebrafish ortholog of K2P 17.1 in comparison to its human counterpart...
May 9, 2018: European Journal of Pharmacology
https://www.readbyqxmd.com/read/29752575/expression-pattern-of-cdkl5-during-zebrafish-early-development-implications-for-use-as-model-for-atypical-rett-syndrome
#6
Marta Vitorino, Nídia Cunha, Natércia Conceição, M Leonor Cancela
Atypical Rett syndrome is a child neurodevelopmental disorder induced by mutations in CDKL5 gene and characterized by a progressive regression in development with loss of purposeful use of the hands, slowed brain and head growth, problems with walking, seizures, and intellectual disability. At the moment, there is no cure for this pathology and little information is available concerning animal models capable of mimicking its phenotypes, thus the development of additional animal models should be of interest to gain more knowledge about the disease...
May 11, 2018: Molecular Biology Reports
https://www.readbyqxmd.com/read/29750281/an-orthotopic-glioblastoma-animal-model-suitable-for-high-throughput-screenings
#7
Linda Pudelko, Steven Edwards, Mirela Balan, Daniel Nyqvist, Jonathan Al-Saadi, Johannes Dittmer, Ingrid Almlöf, Thomas Helleday, Lars Bräutigam
Background: Glioblastoma (GBM) is an aggressive form of brain cancer with poor prognosis. Although murine animal models have given valuable insights into the GBM disease biology, they cannot be used in high-throughput screens to identify and profile novel therapies. The only vertebrate model suitable for large-scale screens, the zebrafish, has proven to faithfully recapitulate biology and pathology of human malignancies and clinically relevant orthotopic zebrafish models have been developed...
May 10, 2018: Neuro-oncology
https://www.readbyqxmd.com/read/29746887/cardiac-ca-2-signalling-in-zebrafish-translation-of-findings-to-man
#8
REVIEW
Chantal J M van Opbergen, Stephanie M van der Voorn, Marc A Vos, Teun P de Boer, Toon A B van Veen
Sudden cardiac death is a leading cause of death worldwide, mainly caused by highly disturbed electrical activation patterns in the heart. Currently, murine models are the most popular model to study underlying molecular mechanisms of inherited or acquired cardiac electrical abnormalities, although the numerous electrophysiological discrepancies between mouse and human raise the question whether mice are the optimal model to study cardiac rhythm disorders. Recently it has been uncovered that the zebrafish cardiac electrophysiology seems surprisingly similar to the human heart, mainly because the zebrafish AP contains a clear plateau phase and ECG characteristics show alignment with the human ECG...
May 7, 2018: Progress in Biophysics and Molecular Biology
https://www.readbyqxmd.com/read/29743679/control-of-cardiac-jelly-dynamics-by-notch1-and-nrg1-defines-the-building-plan-for-trabeculation
#9
Gonzalo Del Monte-Nieto, Mirana Ramialison, Arne A S Adam, Bingruo Wu, Alla Aharonov, Gabriele D'Uva, Lauren M Bourke, Mara E Pitulescu, Hanying Chen, José Luis de la Pompa, Weinian Shou, Ralf H Adams, Sarah K Harten, Eldad Tzahor, Bin Zhou, Richard P Harvey
In vertebrate hearts, the ventricular trabecular myocardium develops as a sponge-like network of cardiomyocytes that is critical for contraction and conduction, ventricular septation, papillary muscle formation and wall thickening through the process of compaction 1 . Defective trabeculation leads to embryonic lethality2-4 or non-compaction cardiomyopathy (NCC) 5 . There are divergent views on when and how trabeculation is initiated in different species. In zebrafish, trabecular cardiomyocytes extrude from compact myocardium 6 , whereas in chicks, chamber wall thickening occurs before overt trabeculation 7 ...
May 9, 2018: Nature
https://www.readbyqxmd.com/read/29741626/functional-analysis-of-a-hypomorphic-allele-shows-that-mmp14-catalytic-activity-is-the-prime-determinant-of-the-winchester-syndrome-phenotype
#10
Ivo J H M de Vos, Evelyn Yaqiong Tao, Sheena Li Ming Ong, Julian L Goggi, Thomas Scerri, Gabrielle R Wilson, Chernis Guai Mun Low, Arnette Shi Wei Wong, Dominic Grussu, Alexander P A Stegmann, Michel van Geel, Renske Janssen, David J Amor, Melanie Bahlo, Norris R Dunn, Thomas J Carney, Paul J Lockhart, Barry J Coull, Maurice A M van Steensel
Winchester syndrome (WS, MIM #277950) is an extremely rare autosomal recessive skeletal dysplasia characterized by progressive joint destruction and osteolysis. To date, only one missense mutation in MMP14, encoding the membrane-bound matrix metalloprotease 14, has been reported in WS patients. Here, we report a novel hypomorphic MMP14 p.Arg111His (R111H) allele, associated with a mitigated form of WS. Functional analysis demonstrated that this mutation, in contrast to previously reported human and murine MMP14 mutations, does not affect MMP14's transport to the cell membrane...
May 8, 2018: Human Molecular Genetics
https://www.readbyqxmd.com/read/29738469/the-role-of-scarnas-in-adjusting-alternative-mrna-splicing-in-heart-development
#11
REVIEW
Chloe Nagasawa, Allison Ogren, Nataliya Kibiryeva, Jennifer Marshall, James E O'Brien, Naoya Kenmochi, Douglas C Bittel
Congenital heart disease (CHD) is a leading cause of death in children <1 year of age. Despite intense effort in the last 10 years, most CHDs (~70%) still have an unknown etiology. Conotruncal based defects, such as Tetralogy of Fallot (TOF), a common complex of devastating heart defects, typically requires surgical intervention in the first year of life. We reported that the noncoding transcriptome in myocardial tissue from children with TOF is characterized by significant variation in levels of expression of noncoding RNAs, and more specifically, a significant reduction in 12 small cajal body-associated RNAs (scaRNAs) in the right ventricle...
May 8, 2018: Journal of Cardiovascular Development and Disease
https://www.readbyqxmd.com/read/29729327/fish-the-better-model-in-human-heart-research-zebrafish-heart-aggregates-as-a-3d-spontaneously-cardiomyogenic-in-vitro-model-system
#12
REVIEW
Patricia Hodgson, Jake Ireland, Bianka Grunow
The zebrafish (ZF) has become an essential model for biomedical, pharmacological and eco-toxicological heart research. Despite the anatomical differences between fish and human hearts, similarities in cellular structure and conservation of genes as well as pathways across vertebrates have led to an increase in the popularity of ZF as a model for human cardiac research. ZF research benefits from an entirely sequenced genome, which allows us to establish and study cardiovascular mutants to better understand cardiovascular diseases...
May 2, 2018: Progress in Biophysics and Molecular Biology
https://www.readbyqxmd.com/read/29727687/variants-in-exosc9-disrupt-the-rna-exosome-and-result-in-cerebellar-atrophy-with-spinal-motor-neuronopathy
#13
David T Burns, Sandra Donkervoort, Juliane S Müller, Ellen Knierim, Diana Bharucha-Goebel, Eissa Ali Faqeih, Stephanie K Bell, Abdullah Y AlFaifi, Dorota Monies, Francisca Millan, Kyle Retterer, Sarah Dyack, Sara MacKay, Susanne Morales-Gonzalez, Michele Giunta, Benjamin Munro, Gavin Hudson, Mena Scavina, Laura Baker, Tara C Massini, Monkol Lek, Ying Hu, Daniel Ezzo, Fowzan S AlKuraya, Peter B Kang, Helen Griffin, A Reghan Foley, Markus Schuelke, Rita Horvath, Carsten G Bönnemann
The exosome is a conserved multi-protein complex that is essential for correct RNA processing. Recessive variants in exosome components EXOSC3, EXOSC8, and RBM7 cause various constellations of pontocerebellar hypoplasia (PCH), spinal muscular atrophy (SMA), and central nervous system demyelination. Here, we report on four unrelated affected individuals with recessive variants in EXOSC9 and the effect of the variants on the function of the RNA exosome in vitro in affected individuals' fibroblasts and skeletal muscle and in vivo in zebrafish...
May 3, 2018: American Journal of Human Genetics
https://www.readbyqxmd.com/read/29725858/zebrafish-heart-failure-models-opportunities-and-challenges
#14
REVIEW
Xingjuan Shi, Ru Chen, Yu Zhang, Junghwa Yun, Koroboshka Brand-Arzamendi, Xiangdong Liu, Xiao-Yan Wen
Heart failure is a complex pathophysiological syndrome of pumping functional failure that results from injury, infection or toxin-induced damage on the myocardium, as well as genetic influence. Gene mutations associated with cardiomyopathies can lead to various pathologies of heart failure. In recent years, zebrafish, Danio rerio, has emerged as an excellent model to study human cardiovascular diseases such as congenital heart defects, cardiomyopathy, and preclinical development of drugs targeting these diseases...
May 3, 2018: Amino Acids
https://www.readbyqxmd.com/read/29723547/hyperglycemia-elicits-anxiety-like-behaviors-in-zebrafish-protective-role-of-dietary-diphenyl-diselenide
#15
Matheus M Dos Santos, Gabriel T de Macedo, Alessandro S Prestes, Vânia L Loro, Graciela M Heidrich, Rochele S Picoloto, Denis B Rosemberg, Nilda V Barbosa
Diabetes mellitus (DM) is a chronic metabolic disease that may comorbid with various psychiatric disorders, such as anxiety and depression. The search for effective therapeutics to alleviate hyperglycemia and complications resulting from DM is continuous. Here we investigate the effects of diphenyl diselenide (DD), an organoselenium compound with several pharmacological properties, in a zebrafish model of hyperglycemia. Fish were fed for 74 days with a diet containing 3 mg/Kg DD, a concentration chosen after experiments based in a dose-response curve (DD 1, 2 and 3 mg/Kg) that did not cause overt toxicity (mortality, weight loss and neurobehavioral deficits)...
April 30, 2018: Progress in Neuro-psychopharmacology & Biological Psychiatry
https://www.readbyqxmd.com/read/29721939/repeatability-and-reproducibility-of-in-vivo-cone-density-measurements-in-the-adult-zebrafish-retina
#16
Alison Huckenpahler, Melissa Wilk, Brian Link, Joseph Carroll, Ross Collery
Zebrafish (Danio rerio) are widely used as an experimental model for a wide range of retinal diseases. Previously, optical coherence tomography (OCT) was introduced for quantitative analysis of the zebrafish cone photoreceptor cell mosaic; however no data exists on the intersession reproducibility or intrasession repeatability of such measurements. We imaged 14 wild-type (WT) fish three times each, with 48 h between each time point. En face images of the UV cone mosaic were generated from the OCT volume scans at each time point...
2018: Advances in Experimental Medicine and Biology
https://www.readbyqxmd.com/read/29707605/scaling-up-to-study-brca2-the-zeppelin-zebrafish-mutant-reveals-a-role-for-brca2-in-embryonic-development-of-kidney-mesoderm
#17
Bridgette E Drummond, Rebecca A Wingert
Specialized renal epithelial cells known as podocytes are essential components of the filtering structures within the kidney that coordinate the process of removing waste from the bloodstream. Podocyte loss initiates many human kidney diseases as it triggers subsequent damage to the kidney, leading to progressive loss of function that culminates with end stage renal failure. Podocyte morphology, function and gene expression profiles are well conserved between zebrafish and humans, making the former a relevant model to study podocyte development and model kidney diseases...
2018: Cancer Cell & Microenvironment
https://www.readbyqxmd.com/read/29696141/enhancing-understanding-of-the-visual-cycle-by-applying-crispr-cas9-gene-editing-in-zebrafish
#18
REVIEW
Rebecca Ward, Husvinee Sundaramurthi, Valeria Di Giacomo, Breandán N Kennedy
During the vertebrate visual cycle, all- trans -retinal is exported from photoreceptors to the adjacent RPE or Müller glia wherein 11- cis -retinal is regenerated. The 11- cis chromophore is returned to photoreceptors, forming light-sensitive visual pigments with opsin GPCRs. Dysfunction of this process perturbs phototransduction because functional visual pigment cannot be generated. Mutations in visual cycle genes can result in monogenic inherited forms of blindness. Though key enzymatic processes are well characterized, questions remain as to the physiological role of visual cycle proteins in different retinal cell types, functional domains of these proteins in retinoid biochemistry and in vivo pathogenesis of disease mutations...
2018: Frontiers in Cell and Developmental Biology
https://www.readbyqxmd.com/read/29692355/bridging-barriers-a-comparative-look-at-the-blood-brain-barrier-across-organisms
#19
REVIEW
Natasha M O'Brown, Sarah J Pfau, Chenghua Gu
The blood-brain barrier (BBB) restricts free access of molecules between the blood and the brain and is essential for regulating the neural microenvironment. Here, we describe how the BBB was initially characterized and how the current field evaluates barrier properties. We next detail the cellular nature of the BBB and discuss both the conservation and variation of BBB function across taxa. Finally, we examine our current understanding of mouse and zebrafish model systems, as we expect that comparison of the BBB across organisms will provide insight into the human BBB under normal physiological conditions and in neurological diseases...
April 1, 2018: Genes & Development
https://www.readbyqxmd.com/read/29684426/mitochondria-targeting-nanomedicine-an-effective-and-potent-strategy-against-aminoglycosides-induced-ototoxicity
#20
Shuang Zhou, Yanhui Sun, Xiao Kuang, Shanshan Hou, YinXian Yang, Zhenjie Wang, Hongzhuo Liu
We report a proof-of-concept for the development of mitochondria-targeting nanoparticles (NPs) loaded with geranylgeranylacetone (GGA) to protect against a wide range of gentamicin-induced ototoxicity symptoms in a zebrafish model. The polymeric NPs were functionalized with a mitochondrial-homing peptide (d‑Arg‑Dmt‑Orn‑Phe‑NH2 ) and exhibited greater mitochondrial uptake and lower gentamicin uptake in hair cells via mechanotransduction (MET) channels and tuned machinery in the hair bundle than the ordinary NPs did...
April 20, 2018: European Journal of Pharmaceutical Sciences
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