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https://www.readbyqxmd.com/read/29228333/dnaaf1-links-heart-laterality-with-the-aaa-atpase-ruvbl1-and-ciliary-intraflagellar-transport
#1
Verity L Hartill, Glenn van de Hoek, Mitali P Patel, Rosie Little, Christopher M Watson, Ian R Berry, Amelia Shoemark, Dina Abdelmottaleb, Emma Parkes, Chiara Bacchelli, Katarzyna Szymanska, Nine V Knoers, Peter J Scambler, Marius Ueffing, Karsten Boldt, Robert Yates, Paul J Winyard, Beryl Adler, Eduardo Moya, Louise Hattingh, Anil Shenoy, Claire Hogg, Eamonn Sheridan, Ronald Roepman, Dominic Norris, Hannah M Mitchison, Rachel H Giles, Colin A Johnson
DNAAF1 (LRRC50) is a cytoplasmic protein required for dynein heavy chain assembly and cilia motility, and DNAAF1 mutations cause primary ciliary dyskinesia (PCD; MIM 613193). We describe four families with DNAAF1 mutations and complex congenital heart disease (CHD). In three families, all affected individuals have typical PCD phenotypes. However, an additional family demonstrates isolated CHD (heterotaxy) in two affected siblings, but no clinical evidence of PCD. We identified a homozygous DNAAF1 missense mutation, p...
December 7, 2017: Human Molecular Genetics
https://www.readbyqxmd.com/read/29225006/mechanistic-studies-on-ketamine-induced-mitochondrial-toxicity-in-zebrafish-embryos
#2
Bonnie L Robinson, Melanie Dumas, Syed F Ali, Merle G Paule, Qiang Gu, Jyotshna Kanungo
Ketamine, a phencyclidine derivative, is an antagonist of the Ca2+-permeable N-methyl-d-aspartate (NMDA)-type glutamate receptors. It is a pediatric anesthetic and has been implicated in developmental neurotoxicity. Ketamine has also been shown to deplete ATP in mammalian cells. Our previous studies showed that acetyl l-carnitine (ALCAR) prevented ketamine-induced cardiotoxicity and neurotoxicity in zebrafish embryos. Based on our finding that ALCAR's protective effect was blunted by oligomycin A, an inhibitor of ATP synthase, we further investigated the effects of ketamine and ALCAR on ATP levels, mitochondria and ATP synthase in zebrafish embryos...
December 7, 2017: Neurotoxicology and Teratology
https://www.readbyqxmd.com/read/29223822/exposure-to-acrylamide-induces-cardiac-developmental-toxicity-in-zebrafish-during-cardiogenesis
#3
Mengmeng Huang, Jingjing Jiao, Jun Wang, Zhidan Xia, Yu Zhang
Acrylamide (AA), an environmental pollutant, has been linked to neurotoxicity, genotoxicity and carcinogenicity. AA is widely used to synthesize polymers for industrial applications, is widely found in Western-style carbohydrate-rich foods and cigarette smoke, and can also be detected in human umbilical cord blood and breast milk. This is the first study that demonstrated the cardiac developmental toxicity of AA in zebrafish embryos. Post-fertilization exposure to AA caused a clearly deficient cardiovascular system with a shrunken heart and abortive morphogenesis and function...
December 7, 2017: Environmental Pollution
https://www.readbyqxmd.com/read/29221954/total-particulate-matter-from-cigarette-smoke-disrupts-vascular-development-in-zebrafish-brain-danio-rerio
#4
Andrey Massarsky, G L Prasad, Richard T Di Giulio
Several studies have demonstrated zebrafish as a useful high-throughput in-vivo model to study the effects of cigarette smoke on early development. It has been shown previously that exposure of zebrafish to cigarette smoke total particulate matter (TPM) leads to several adverse physiological aberrations, including heart deformities and improper angiogenesis. Consequently, this study investigated the effects of TPM on cardiovascular development in zebrafish that were exposed to increasing concentrations of TPM based upon nicotine content from 6h post fertilization (hpf) up to 72hpf...
December 5, 2017: Toxicology and Applied Pharmacology
https://www.readbyqxmd.com/read/29203887/age-dependent-increase-of-oxidative-stress-regulates-microrna-29-family-preserving-cardiac-health
#5
Johanna Heid, Chiara Cencioni, Roberto Ripa, Mario Baumgart, Sandra Atlante, Giuseppina Milano, Alessandro Scopece, Carsten Kuenne, Stefan Guenther, Valerio Azzimato, Antonella Farsetti, Giacomo Rossi, Thomas Braun, Giulio Pompilio, Fabio Martelli, Andreas M Zeiher, Alessandro Cellerino, Carlo Gaetano, Francesco Spallotta
The short-lived turquoise killifish Nothobranchius furzeri (Nfu) is a valid model for aging studies. Here, we investigated its age-associated cardiac function. We observed oxidative stress accumulation and an engagement of microRNAs (miRNAs) in the aging heart. MiRNA-sequencing of 5 week (young), 12-21 week (adult) and 28-40 week (old) Nfu hearts revealed 23 up-regulated and 18 down-regulated miRNAs with age. MiR-29 family turned out as one of the most up-regulated miRNAs during aging. MiR-29 family increase induces a decrease of known targets like collagens and DNA methyl transferases (DNMTs) paralleled by 5´methyl-cytosine (5mC) level decrease...
December 4, 2017: Scientific Reports
https://www.readbyqxmd.com/read/29202458/integrating-light-sheet-imaging-with-virtual-reality-to-recapitulate-developmental-cardiac-mechanics
#6
Yichen Ding, Arash Abiri, Parinaz Abiri, Shuoran Li, Chih-Chiang Chang, Kyung In Baek, Jeffrey J Hsu, Elias Sideris, Yilei Li, Juhyun Lee, Tatiana Segura, Thao P Nguyen, Alexander Bui, René R Sevag Packard, Peng Fei, Tzung K Hsiai
Currently, there is a limited ability to interactively study developmental cardiac mechanics and physiology. We therefore combined light-sheet fluorescence microscopy (LSFM) with virtual reality (VR) to provide a hybrid platform for 3D architecture and time-dependent cardiac contractile function characterization. By taking advantage of the rapid acquisition, high axial resolution, low phototoxicity, and high fidelity in 3D and 4D (3D spatial + 1D time or spectra), this VR-LSFM hybrid methodology enables interactive visualization and quantification otherwise not available by conventional methods, such as routine optical microscopes...
November 16, 2017: JCI Insight
https://www.readbyqxmd.com/read/29201567/the-zebrafish-as-a-model-system-for-analyzing-mammalian-and-native-%C3%AE-crystallin-promoter-function
#7
Mason Posner, Kelly L Murray, Matthew S McDonald, Hayden Eighinger, Brandon Andrew, Amy Drossman, Zachary Haley, Justin Nussbaum, Larry L David, Kirsten J Lampi
Previous studies have used the zebrafish to investigate the biology of lens crystallin proteins and their roles in development and disease. However, little is known about zebrafish α-crystallin promoter function, how it compares to that of mammals, or whether mammalian α-crystallin promoter activity can be assessed using zebrafish embryos. We injected a variety of α-crystallin promoter fragments from each species combined with the coding sequence for green fluorescent protein (GFP) into zebrafish zygotes to determine the resulting spatiotemporal expression patterns in the developing embryo...
2017: PeerJ
https://www.readbyqxmd.com/read/29197232/uptake-tissue-distribution-and-toxicity-of-polystyrene-nanoparticles-in-developing-zebrafish-danio-rerio
#8
Jordan A Pitt, Jordan S Kozal, Nishad Jayasundara, Andrey Massarsky, Rafael Trevisan, Nick Geitner, Mark Wiesner, Edward D Levin, Richard T Di Giulio
Plastic pollution is a critical environmental concern and comprises the majority of anthropogenic debris in the ocean, including macro, micro, and likely nanoscale (less than 100nm in at least one dimension) plastic particles. While the toxicity of macroplastics and microplastics is relatively well studied, the toxicity of nanoplastics is largely uncharacterized. Here, fluorescent polystyrene nanoparticles (PS NPs) were used to investigate the potential toxicity of nanoplastics in developing zebrafish (Danio rerio), as well as to characterize the uptake and distribution of the particles within embryos and larvae...
November 24, 2017: Aquatic Toxicology
https://www.readbyqxmd.com/read/29196619/opposite-effects-of-activin-type-2-receptor-ligands-on-cardiomyocyte-proliferation-during-development-and-repair
#9
Deepika Dogra, Suchit Ahuja, Hyun-Taek Kim, S Javad Rasouli, Didier Y R Stainier, Sven Reischauer
Zebrafish regenerate damaged myocardial tissue very effectively. Hence, insights into the molecular networks underlying zebrafish heart regeneration might help develop alternative strategies to restore human cardiac performance. While TGF-β signaling has been implicated in zebrafish cardiac regeneration, the role of its individual ligands remains unclear. Here, we report the opposing expression response during zebrafish heart regeneration of two genes, mstnb and inhbaa, which encode TGF-β family ligands...
December 1, 2017: Nature Communications
https://www.readbyqxmd.com/read/29194605/how-the-expression-of-green-fluorescent-protein-and-human-cardiac-actin-in-the-heart-influences-cardiac-function-and-aerobic-performance-in-zebrafish-danio-rerio
#10
S R Avey, M Ojehomon, J F Dawson, T E Gillis
The present study examined how the expression of enhanced green fluorescent protein (eGFP) and human cardiac actin (ACTC) in zebrafish Danio rerio influences embryonic heart rate (RH ) and the swim performance and metabolic rate of adult fish. Experiments with the adults involved determining the critical swimming speed (Ucrit , the highest speed sustainable and measure of aerobic capacity) while measuring oxygen consumption. Two different transgenic D. rerio lines were examined: one expressed eGFP in the heart (tg(cmlc:egfp)), while the second expressed ACTC in the heart and eGFP throughout the body (tg(cmlc:actc,ba:egfp))...
November 30, 2017: Journal of Fish Biology
https://www.readbyqxmd.com/read/29194345/embryonic-ethanol-exposure-affects-early-and-late-added-cardiac-precursors-and-produces-long-lasting-heart-chamber-defects-in-zebrafish
#11
Swapnalee Sarmah, James A Marrs
Drinking mothers expose their fetuses to ethanol, which produces birth defects: craniofacial defects, cognitive impairment, sensorimotor disabilities and organ deformities, collectively termed as fetal alcohol spectrum disorder (FASD). Various congenital heart defects (CHDs) are present in FASD patients, but the mechanisms of alcohol-induced cardiogenesis defects are not completely understood. This study utilized zebrafish embryos and older larvae to understand FASD-associated CHDs. Ethanol-induced cardiac chamber defects initiated during embryonic cardiogenesis persisted in later zebrafish life...
December 1, 2017: Toxics
https://www.readbyqxmd.com/read/29193635/inactivation-of-ammecr1-is-associated-with-growth-bone-and-heart-alterations
#12
Mariana Moysés-Oliveira, Giuliana Giannuzzi, Richard J Fish, Jill A Rosenfeld, Florence Petit, Maria de Fatima Soares, Leslie Domenici Kulikowski, Adriana Di Battista, Malú Zamariolli, Fan Xia, Thomas Liehr, Nadezda Kosyakova, Gianna Carvalheira, Michael Parker, Eleanor G Seaby, Sarah Ennis, Rodney D Gilbert, R Tanner Hagelstrom, Maria L Cremona, Wenhui L Li, Alka Malhotra, Anjana Chandrasekhar, Denise L Perry, Ryan J Taft, Julie McCarrier, Donald G Basel, Joris Andrieux, Taiza Stumpp, Fernanda Antunes, Gustavo José Pereira, Marguerite Neerman-Arbez, Vera Ayres Meloni, Margaret Drummond-Borg, Maria Isabel Melaragno, Alexandre Reymond
We report five individuals with loss-of-function of the X-linked AMMECR1: a girl with a balanced X-autosome translocation and inactivation of the normal X-chromosome; two boys with maternally-inherited and de novo nonsense variants; and two half-brothers with maternally inherited microdeletion variants. They present with short stature, cardiac and skeletal abnormalities and hearing loss. Variants of unknown significance in AMMECR1 in four male patients from two families with partially overlapping phenotypes were previously reported...
November 28, 2017: Human Mutation
https://www.readbyqxmd.com/read/29190094/active-glucocorticoids-have-a-range-of-important-adverse-developmental-and-physiological-effects-on-developing-zebrafish-embryos
#13
Raffael Alois Willi, Susanne Faltermann, Timm Hettich, Karl Fent
Glucocorticoids in aquatic systems originating from natural excretion and medical use may pose a risk to fish. Here, we analyzed physiological and transcriptional effects of clobetasol propionate (CLO), cortisol and cortisone in zebrafish embryos as single compounds and binary mixtures. CLO and cortisol, but not cortisone showed a concentration-dependent decrease in muscle contraction, increase in heart beating, and accelerated hatching. CLO also induced immobilization and edema at high concentrations. Transcription analysis covering up to 26 genes showed that mostly genes related to glucose metabolism, immune system and development were differentially expressed at 91 ng/L and higher...
November 30, 2017: Environmental Science & Technology
https://www.readbyqxmd.com/read/29184141/development-and-growth-of-organs-in-living-whole-embryo-and-larval-grafts-in-zebrafish
#14
Toshihiro Kawasaki, Akiteru Maeno, Toshihiko Shiroishi, Noriyoshi Sakai
Age-related systemic environments influence neurogenesis and organ regeneration of heterochronic parabiotic partners; however, the difficulty of manipulating small embryos prevents the effects of aged systemic environments on primitive organs at the developmental stage from being analysed. Here, we describe a novel transplantation system to support whole living embryos/larvae as grafts in immunodeficient zebrafish by the intrusion of host blood vessels into the grafts, allowing bodies similar to those of heterochronic parabiosis to be generated by subcutaneous grafting...
November 28, 2017: Scientific Reports
https://www.readbyqxmd.com/read/29183943/anisotropic-shear-stress-patterns-predict-the-orientation-of-convergent-tissue-movements-in-the-embryonic-heart
#15
Francesco Boselli, Emily Steed, Jonathan B Freund, Julien Vermot
Myocardial contractility and blood flow provide essential mechanical cues for the morphogenesis of the heart. In general, endothelial cells change their migratory behavior in response to shear stress patterns, according to flow directionality. Here, we assessed the impact of shear stress patterns and flow directionality on the behavior of endocardial cells, the specialized endothelial cells of the heart. At the early stages of zebrafish heart valve formation, we show that endocardial cells are converging to the valve-forming area and that this behavior depends upon mechanical forces...
December 1, 2017: Development
https://www.readbyqxmd.com/read/29176804/one-crisis-diverse-impacts-tissue-specificity-of-folate-deficiency-induced-circulation-defects-in-zebrafish-larvae
#16
Hung-Chi Tu, Gang-Hui Lee, Tsun-Hsien Hsiao, Tseng-Ting Kao, Tzu-Ya Wang, Jen-Ning Tsai, Tzu-Fun Fu
Folate (vitamin B9) is an essential nutrient required for cell survival, proliferation, differentiation and therefore embryogenesis. Folate deficiency has been associated with many diseases, including congenital heart diseases and megaloblastic anemia, yet the mechanisms underlying these remains elusive. Here, we examine the impact of folate deficiency on the development of the circulation system using a zebrafish transgenic line which displays inducible folate deficiency. Impaired hematopoiesis includes decreased hemoglobin levels, decreased erythrocyte number, increased erythrocyte size and aberrant c-myb expression pattern were observed in folate deficient embryos...
2017: PloS One
https://www.readbyqxmd.com/read/29174768/acetylation-of-tbx5-by-kat2b-and-kat2a-regulates-heart-and-limb-development
#17
Tushar K Ghosh, José J Aparicio-Sánchez, Sarah Buxton, Ami Ketley, Tasabeeh Mohamed, Catrin S Rutland, Siobhan Loughna, J David Brook
TBX5 plays a critical role in heart and forelimb development. Mutations in TBX5 cause Holt-Oram syndrome, an autosomal dominant condition that affects the formation of the heart and upper-limb. Several studies have provided significant insight into the role of TBX5 in cardiogenesis; however, how TBX5 activity is regulated by other factors is still unknown. Here we report that histone acetyltransferases KAT2A and KAT2B associate with TBX5 and acetylate it at Lys339. Acetylation potentiates its transcriptional activity and is required for nuclear retention...
November 22, 2017: Journal of Molecular and Cellular Cardiology
https://www.readbyqxmd.com/read/29162723/the-transcription-factor-foxc1a-in-zebrafish-directly-regulates-expression-of-nkx2-5-encoding-a-transcriptional-regulator-of-cardiac-progenitor-cells
#18
Yunyun Yue, Mingyang Jiang, Luqingqing He, Zhaojunjie Zhang, Qinxin Zhang, Chun Gu, Meijing Liu, Nan Li, Qingshun Zhao
Cardiogenesis is a tightly controlled biological process required for formation of a functional heart. The transcription factor Foxc1 not only plays a crucial role in outflow tract development in mice, but is also involved in  cardiac structure formation and normal function in human. However, the molecular mechanisms by which Foxc1 controls cardiac development remain poorly understood. Previously, we reported that zebrafish embryos deficient in foxc1a, an ortholog of mammalian Foxc1, display pericardial edemas and die 9-10 days post fertilization...
November 21, 2017: Journal of Biological Chemistry
https://www.readbyqxmd.com/read/29162721/loss-of-%C3%AE-b-crystallin-function-in-zebrafish-reveals-critical-roles-in-the-development-of-the-lens-and-stress-resistance-of-the-heart
#19
Sanjay Mishra, Shu-Yu Wu, Alexandra W Fuller, Zhen Wang, Kristie L Rose, Kevin L Schey, Hassane S Mchaourab
Genetic mutations in the human small heat shock protein αB-crystallin have been implicated in autosomal cataracts and skeletal myopathies, including heart muscle diseases (cardiomyopathy). While these mutations lead to modulation of their chaperone activity in vitro, the in vivo functions of αB-crystallin in the maintenance of both lens transparency and muscle integrity remain unclear. This lack of information has hindered a mechanistic understanding of these diseases. To better define the functional roles of αB-crystallin, we generated loss-of-function zebrafish mutant lines by utilizing CRISPR/Cas9 system to specifically disrupt the two αB-crystallin genes, αBa and αBb We observed lens abnormalities in the mutant lines of both genes and the penetrance of the lens phenotype was higher in αBa than αBb mutants...
November 21, 2017: Journal of Biological Chemistry
https://www.readbyqxmd.com/read/29161412/fluid-dynamics-in-heart-development-effects-of-hematocrit-and-trabeculation
#20
Nicholas A Battista, Andrea N Lane, Jiandong Liu, Laura A Miller
Recent in vivo experiments have illustrated the importance of understanding the haemodynamics of heart morphogenesis. In particular, ventricular trabeculation is governed by a delicate interaction between haemodynamic forces, myocardial activity, and morphogen gradients, all of which are coupled to genetic regulatory networks. The underlying haemodynamics at the stage of development in which the trabeculae form is particularly complex, given the balance between inertial and viscous forces. Small perturbations in the geometry, scale, and steadiness of the flow can lead to changes in the overall flow structures and chemical morphogen gradients, including the local direction of flow, the transport of morphogens, and the formation of vortices...
November 16, 2017: Mathematical Medicine and Biology: a Journal of the IMA
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