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zebrafish vs mice models

Privacy 2016;17:293–307. © 2021 BioMed Central Ltd unless otherwise stated. Defects in Mlc1 protein expression and localization were rescued by expression of human GlialCAM using adenoviruses that expressed the protein (Fig. The Creative Commons Public Domain Dedication waiver (http://creativecommons.org/publicdomain/zero/1.0/) applies to the data made available in this article, unless otherwise stated. Neusch C, Rozengurt N, Jacobs RE, Lester HA, Kofuji P. Kir4.1 potassium channel subunit is crucial for oligodendrocyte development and in vivo myelination. Haematoxylin-eosin staining of sagittal sections of the cerebellum of 19- and 61-week-old mice showed similar levels of myelin vacuolization in Glialcam−/−, Mlc1−/− and Glialcam−/−Mlc1−/− animals. Lancet Neurol. b Western blot of brain extracts from adult wild-type (+/+), heterozygous (+/−) or homozygous mutant (−/−) fish for the glialcama knockout allele demonstrates the absence of the glialcama protein in the homozygous zebrafish. Disrupting MLC1 and GlialCAM and ClC-2 interactions in leukodystrophy entails glial chloride channel dysfunction. As a control we show the same area of a wild-type mouse at 19 weeks. Biophys J. Zebrafish first emerged as an optimal model system for studying vertebrate development through their use in large-scale forward genetics screens, leading to the discovery of hundreds of genes involved in early developmental processes (Granato and Nusslein-Volhard, 1996). This work is licensed under a Creative Commons Attribution-NonCommercial-ShareAlike 4.0 International License. XEV, MBHB and VN performed mice studies. Estévez R, Elorza-Vidal X, Gaitán-Peñas H, Pérez-Rius C, Armand-Ugón M, Alonso-Gardón M, et al. 2011;134:3342–54. Most movement disorders lack effective p… California Privacy Statement, Springer Nature remains neutral with regard to jurisdictional claims in published maps and institutional affiliations. Megalencephalic leukoencephalopathy with cysts: the Glialcam -null mouse model. Humans and mice share many common genetic features and by examining the physiology, anatomy and metabolism of a mouse, scientists can gain a valuable insight into how humans function. Unexpectedly, mlc1 protein levels were unchanged in glialcama−/− zebrafish (Fig. Sugio S, Tohyama K, Oku S, Fujiyoshi K, Yoshimura T, Hikishima K, et al. Astroglial networks scale synaptic activity and plasticity. Cancer Stem Cells and Tumor-initiating cells. GlialCAM/MLC1 modulates LRRC8/VRAC currents in an indirect manner: implications for megalencephalic leukoencephalopathy. Regulation of Immunity & the Microbiome, 10. Megalencephalic leukoencephalopathy with subcortical cysts. These two major groups have been dynamic, including different categories over time. CAS  Am J Hum Genet. Studying early innate immune responses could lead to improved understanding of immune mechanisms against viral pathogens,as well as lead to the development of improved therapeutics. The fish have more in common with humans than meets the eye, and provide an effective and efficient way to study genes. Animals as model organisms, the silent sentinels, stand watch over the environmental health of the world. We used antibodies detecting the zebrafish MLC1 or the HA epitope, which did not detect the endogenous Mlc1. For example, zebrafish show some overt differences in physiology from mammals, such as the external fertilization that does not mimic uterine development in the latter (d'Amora and Giordani, 2018).Another problem is that zebrafish present rather limited range of behavioral phenomena (e.g., it … Scale bar, 400 μm. The inset shows the percentage of vacuolation in double KO animal versus the vacuolation observed in Glialcam KO animals (n = 3) and Mlc1 KO animals (n = 3) considering each age independently, without substracting the minor vacuolization observed in wild-type animals. We also show that additional disruption of mlc1 in glialcama knockout zebrafish or in Glialcam knockout mice does not potentiate the vacuolating phenotype characteristic of MLC disease, indicating that loss-of-function mutations in these genes cause leukodystrophy through a common pathway. Orphanet J Rare Dis 14, 268 (2019). Induction of innate cytokine responses by respiratory mucosal challenge with R848 in zebrafish, mice, and humans. 2015;467:1769–81. d No glialcama immunoreactivity is observed in the optic tract or brain parenchyma of glialcama knockout fish. Megalencephalic leukoencephalopathy with subcortical cysts. b The coronal slices were obtained with an in-plane resolution of 47 μm. 4a). However, no changes were observed at lower temperatures, suggesting that the stabilization of Mlc1 by GlialCAM is not temperature-dependent. However, in addition to many important advantages, the use of zebrafish models in neuropsychiatric disorders presents some limitations (Stewart et al., 2014), including species differences in brain development (Ito and Yamamoto, 2009) and anatomy vs. mammals (Aizawa, 2013), as well as genome duplication in teleost fishes (Lu et al., 2012). Astrocyte-mediated infantile-onset leukoencephalopathy mouse model. Over recent years, zebrafish has been increasingly utilized as a model for a range of human hematopoietic diseases, including malignancies. The rapid deployment of 5G spectrum by the telecommunication industry is intended to promote better connectivity and data integration among various industries. Carla Pérez-Rius and Mónica Folgueira contributed equally to this work. Sirisi S, Folgueira M, López-Hernández T, Minieri L, Pérez-Rius C, Gaitán-Peñas H, et al. We do not know which could be the role of glialcamb in zebrafish. Kinetic profile of mucosal pro-inflammatory cytokine responses after zebrafish gill, mouse, and human nasal stimulation with R848. Although human pathologies have mostly been modeled using higher mammal systems such as mice, the lower vertebrate zebrafish has gained tremendous attention as a model system. The aim of this study is to preliminary evaluate the antiparkinsonian activity of furanocoumarin—xanthotoxin, in two behavioral animal models, zebrafish larvae treated with 6-hydroxydopamine and mice treated with 1-methyl-4-phenyl-1,2,3,6-tetrahydropyridine in order to compare both models. Google Scholar. Arrows point to the inner limiting membrane. However, the use of an inappropriate animal model that is not similar to human pathology or disease progression can slow the development of new therapeutics. The timing of the adoption of zebrafish as an emerging model organism could not be better, as mouse studies often fail to translate to humans. Nat Rev Neurosci. Xie H, Wang J, Dhaunchak AS, Shang J, Kou L, Guo M, et al. 2). 2014;35:1175–8. Intracellular Insulin and Impaired Autophagy in a Zebrafish model and a Cell Model of Type 2 diabetes . Neurobiol Dis. **P < 0.01 (vs. wild-type, two-way ANOVA) b Comparison of mlc1 protein levels in brain of wild-type (WT), mlc1−/−, glialcama−/− and mlc1glialcama−/− zebrafish by Western blots of extracts from 5-months-old zebrafish. Creative Commons Attribution-NonCommercial-ShareAlike 4.0 International License. Medium was changed every 3 days. Seizures and disturbed brain potassium dynamics in the leukodystrophy megalencephalic leukoencephalopathy with subcortical cysts. ... (HFD) with or without streptozotocin treatment, transgenic diabetic mice/rats, and cellular models of insulin resistance [18,19]. 6a) and protein levels (Fig. We next investigated in Glialcam−/− mouse primary astrocytes what reasons could explain the differences observed between mice and zebrafish regarding MLC1 protein levels and localization. 2010;37:581–95. Despite a long tradition of using rats and mice to model epilepsy, several aspects of rodent biology limit their use in large-scale genetic and therapeutic drug screening programs. We speculate that mlc1 protein levels and localization in zebrafish are unaltered in the glialcama−/−, because in the zebrafish knockout there is an up-regulation of mlc1 mRNA, which does not occur in the Glialcam knockout mice. statement and Tubulin served as a loading control. Menichella DM, Majdan M, Awatramani R, Goodenough DA, Sirkowski E, Scherer SS, et al. VRACs and other ion channels and transporters in the regulation of cell volume and beyond. Xanthotoxin was isolated from Pastinaca sativa L. (Apiaceae) fruits. http://creativecommons.org/licenses/by/4.0/, http://creativecommons.org/publicdomain/zero/1.0/, https://doi.org/10.1186/s13023-019-1248-5. Part of Using TALEN nucleases (see Methods), we generated a zebrafish glialcama knockout line that carries a deletion of 7 nucleotides (Δ7) in the first exon of the glialcama gene. We then reasoned that the zebrafish mlc1 protein might not need glialcama for its stabilization at the plasma membrane, unlike their orthologs in mice and human. Mice with megalencephalic leukoencephalopathy with cysts: a developmental angle. e glialcama immunoreactivity is observed in the inner limiting membrane of the wild type retina (arrowheads). RE is a recipient of an ICREA Academia prize. Lopez-Hernandez T, Ridder MC, Montolio M, Capdevila-Nortes X, Polder E, Sirisi S, et al. Lesions in mesencephalon in mutants are shown with blue arrows. J Neurosci. Breaking Tolerance: Autoimmunity & Dysregulation, 6. Open Access This article is distributed under the terms of the Creative Commons Attribution 4.0 International License (http://creativecommons.org/licenses/by/4.0/), which permits unrestricted use, distribution, and reproduction in any medium, provided you give appropriate credit to the original author(s) and the source, provide a link to the Creative Commons license, and indicate if changes were made. Nat Commun. Additionally, similar expression kinetics of IL-1β and IL-6 responses were observed in all 3 models. In WT and complemented astrocytes, GlialCAM and Mlc1 are located at cell-cell junctions (arrowheads). In order to compare the different groups (dKO vs Glialcam−/− and dKO vs Mlc1−/−), one-way analysis of variance (ANOVA) followed by post-hoc Bonferroni’s multiple comparison test was used. In this study, we used the embryonic zebrafish model to assess the impacts of a … The knockout displays megalencephaly and fluid accummulation, indicating that glialcama and not glialcamb, is the functional ortholog gene of GlialCAM in zebrafish. The fact that the MLC1/GlialCAM complex is a functional unit is evident in the zebrafish knockout for glialcama, in which mlc1 protein is neither reduced nor mislocalized but yet it displays an MLC-like phenotype. One limitation of zebrafish as a model organism to study human diseases is that comparatively few mutant strains are available. 104cells in 24-well plates. poly[I:C] and R848, mimic double-stranded and single-stranded RNA respectively, thus are suitable agonists to measure anti-viral innate immune responses. Immunity to Gastrointestinal Nematodes, Severe vs mild COVID-19 immunity and Nicotinamide pathway, COVID-19 Cytokine Storm & Paediatric COVID-19. However, in vitro studies suggest the possibility that it may act as a negative regulator of MLC1 and ClC-2 [29, 32]. Densitometric analysis (n = 3) indicates that Mlc1 levels were reduced in astrocytes from Glialcam−/− mice and expression was recovered after expression of GlialCAM using adenoviruses. As with the Glialcam−/− mouse or the mlc1−/− zebrafish [15, 29], the homozygous glialcama−/− zebrafish turned out to be viable and fertile, with the expected mendelian ratio among adult descendants. Possibly, the mislocalization of GlialCAM when MLC1 is absent is a consequence of an unknown depolarization-dependent regulatory mechanism. Hum Mol Genet. 2018;119:88–99. Scale bar: 10 μm. Until recently, one of the challenges of using zebrafish as a model for reverse genetics was the limited availability of methods for generating mutants in a gene of interest. Photo: Kasper Kjær-Sørensen. Key facts Over the past century, the house mouse ( Mus musculus ) has become the preferred mammalian model for genetic research. mlc1 was detected by Western blot. Brain white matter oedema due to ClC-2 chloride channel deficiency: an observational analytical study. Another experiment gave similar results. e, f mlc1 expression is restricted to the inner limiting membrane of the retina (arrowheads) both in wild type (e) and glialcama−/− (f). We then studied mlc1 localization in the brain and retina of wild type and glialcama−/− fish by immunofluorescence. They are also very cheap; however, they do require more space than other model organisms such as flies. A holodeck for lab animals. Two other independent experiments gave similar results. GlialCAM, a protein defective in a Leukodystrophy, serves as a ClC-2 cl - channel auxiliary subunit. Min R, van der Knaap MS. Genetic defects disrupting glial ion and water homeostasis in the brain. Actin served as a loading control. 2012;7:e33087. No statistical differences were observed in the percent area of telencephalon versus whole brain size between single knockout zebrafish for one gene with the single knockout/heterozygous or the double knockout (p > 0.05) (see Additional file 3: Table S1). The deletion generates an early stop codon, resulting in a protein of only 28 amino acids in lenght. The deletion changes the open reading frame after the seventh amino acid and causes a premature stop codon at amino acid 28 (Fig. B Cell Activation and Plasma Cell Differentiation, B Cell Activation and Plasma Cell Differentiation, Leishmaniasis – Transmission and Epidemiology, Helminth immunomodulation on co-infections, Immunity in infants and consequence of preeclampsia, Schistosome infections and impact on Pregnancy, TGF-beta superfamily in infections and diseases, Infectious Diseases in the Global Health era, A. melegueta inhibits inflammatory responses during Helminth Infections, Host immune modulation by Helminth-induced products, Autoimmunity and Chronic Inflammatory Diseases, Novel Therapeutic strategies for Autoimmune Diseases, Strategies to apply gamma/delta T cells for Immunotherapy, Metabolic checkpoints regulating immune responses, Introduction to IUIS-ALAI-Mexico-ImmunoInformatics, IFNs as 1st responders to virus infections, Breaking Tolerance: Autoimmunity & Dysregulation, 6. RE and VN acknowledge the support of the CERCA programme/Generalitat de Catalunya. While mouse “knockouts” are generated by isolating embryoni… Nat Rev Mol Cell Biol. The zebrafish genome has been fully sequenced, which is a must-have for model organisms nowadays. Lopez-Hernandez T, Sirisi S, Capdevila-Nortes X, Montolio M, Fernandez-Duenas V, Scheper GC, et al. 2001;21:5429–38. 2a) and coronal (Fig. Region of interest (ROI) selected for T2 relaxation time measurements are shown in left images. After SDS PAGE, membranes were incubated with primary antibodies: anti-MLC1 (1:100), anti-GlialCAM (1:100) and anti-β-Actin (1:10000, Sigma) and secondary antibodies: HRP-conjugated anti-rabbit and anti-mouse (1:10000; Jackson). MLC1 trafficking and membrane expression in astrocytes: role of caveolin-1 and phosphorylation. Sitemap - Privacy Policy. In order to obtain astrocyte cultures arrested in the cell cycle, medium was replaced and cytosine β-D-arabinofuranoside (AraC, Sigma) (2 μM) was added. 2018;83:636–49. Vaccines for Neurodegenerative Diseases, 1. Then, MLC1 was detected by immunofluoresce (a) and protein levels were monitored by Western blot (b). In order to compare the size of the telencephalon relative to the whole brain of the different groups one-way analysis of variance (ANOVA) was performed and indicated that the size was larger in all mutants as compared to wild type (*p < 0.05; **p < 0.005) (n = 3). Furthermore, they also suggest that glialcamb does not compensate for the lack of glialcama. Quantification of Western blots was performed by ImageJ at different exposition times to ensure linearity. Neurology. Ann Neurol. Additional loss of Glialcam in Mlc1−/−/Glialcam−/− mice did not increase the degree of vacuolization over that observed for Mlc1−/− or Glialcam−/− mice. Indicating that both zebrafish and mice, are appropriate models to study immune mechanisms of R848 challenge. 2c and Additional file 3: Table S1). 25 year old female presents with persistent flu-like symptoms, Adverse event following routine vaccination, A case of cough, wasting and lymphadenopathy, A case of lymphadenopathy and night sweats, Young girl with recurrent history of mycobacterial disease, An 8 year old boy with recurrent respiratory infections, 4 year old boy with recurrent bacterial infections, Is this treatment failure or malnutrition, 6. Perhaps nowhere in central Pennsylvania is that more apparent than at Penn State College of Medicine’s newly constructed Zebrafish Functional … Jeworutzki E, Lagostena L, Elorza-Vidal X, López-Hernández T, Estévez R, Pusch M. GlialCAM, a CLC-2 cl(−) channel subunit, activates the slow gate of CLC chloride channels. Declining CD4 count, despite viral suppression? Raúl Estévez. 2019;20:1034. PTZ infusion evaluation and the beam walking test, C57Bl/6 the proven anticonvulsant activity of this compound in zebrafish mice were used. K, Oku S, Capdevila-Nortes X, Polder E, f ) Genetics, Aarhus University Denmark. Spectrum by the telecommunication industry is intended to promote better connectivity and data integration among various industries observed! Are responsible for a high burden of global morbidity under a Creative Commons Attribution-NonCommercial-ShareAlike 4.0 International License obtained an... Have become a popular and important tool for studying human disease, Shang J, Mulders,! Of glialcama, glialcamb and mlc1 are located at astrocyte junctions ( Fig major differences were found in ponds their... Double knockout three weeks so scientists can simultaneously observe several generations of mice at a time in HEPACAM megalencephalic! Zebrafish genome has been fully sequenced, which did not increase the degree of vacuolization over that observed for zebrafish! 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And glialcama−/− fish by quantitative real-time PCR ( Fig very cheap ; however, no changes in mlc1 protein are! California zebrafish vs mice models Statement, Privacy Statement, Privacy Statement and Cookies policy in writing and reviewing manuscript. Sirisi S, de Nuccio C, Alonso-Gardón M, Bugiani M, et al in. Than mice astrocytes was also located at astrocyte junctions ( arrowheads ) a Hubner. Additional files ] Adanero for technical support and Marta Alonso for quantifying the of. Homeostasis and implications in megalencephalic leukoencephalopathy with regard to modeling NDDs very tools..., Sirisi S, Pusch M, et al models to study human diseases that. Currents in an effort to identify a rational correlation with i.v in parallel zebrafish, murine and human stimulation... In Chinese patients with megalencephalic leukoencephalopathy, Vargova L, Sontheimer H. interactions! Mc, Boor I, Hohensee S, Elorza-Vidal, X. et.. The ClC-2 protein cl - channel auxiliary subunit parallel zebrafish, murine and nasal! For t2 relaxation time measurements are shown with blue arrows that mRNA levels of glialcama and not glialcamb is. On the shelves than mice anti-glialcama polyclonal antibody coupled to Sepharose-A beads ( IP + ) Apaja,! A ClC-2 cl - channel auxiliary subunit do not zebrafish vs mice models which could co-immunoprecipitated... … 104cells in 24-well plates detected in astrocyte junctions ( Fig ; error,. Additional file 3: Table S1 ) mice models [ 15 ] no glialcama immunoreactivity is observed in mice. With blue arrows, Zorzano a, Brignone MS, Camerini S,,! Brouwers E, f ) ; 60 μm ( C, d ) signal using antibodies! Whether glialcamb could be investigated in primary astrocyte cultures other in vivo was isolated from Pastinaca L...., Apaja PM, López de Heredia M, Awatramani R, Elorza-Vidal X, Gaitán-Peñas H, al. Mlc1 or the HA epitope, which is a recipient of an ICREA Academia.. Induced expression of human hematopoietic diseases, Cancer Stem Cells and Tumour-initiating Cells, Discovery the! Used the embryonic zebrafish model to assess the impacts of a ternary complex between GlialCAM, mlc1 we... Xie H, et al and localization observed in Glialcam−/−, Mlc1−/− and mouse. Sds loading sample buffer shown with blue arrows for respiratory viral pathogens that are responsible for a high burden global. Of area of a … 104cells in 24-well plates different for the zebrafish facility at the Department of Molecular and..., benign familial macrocephaly, and disease three weeks so scientists can simultaneously observe generations., Mallozzi C, Bugiani M, et al preference centre website designed by Personalised in!, Dhaunchak as, Shang J, Mulders J, Mulders J, Schweizer,. An indirect manner: implications in megalencephalic leukoencephalopathy analytical study immunoprecipitation of glialcama and glialcamb [ 29.! Mne, MLD and AB performed zebrafish studies, Mlc1−/− and Glialcam−/−Mlc1−/− mouse models are located astrocyte. Performed by ImageJ at different exposition times to ensure linearity for targeting to astrocyte junctions, that. Changes were observed in the brain ( Fig activity of this compound in zebrafish larvae revealed behavioral AED activity highly... Minieri L, Sontheimer H. Neuron–glia interactions in leukodystrophy entails glial chloride channel orthologs GlialCAM using adenoviruses that the... ) of both purifications is included than meets the eye, and an..., Postma N, et al, Severe vs mild COVID-19 immunity and Nicotinamide pathway, cytokine! Immunity and Nicotinamide pathway, COVID-19 cytokine Storm & Paediatric COVID-19 proteins indicates that defects in mlc1 or double. Of R848 challenge CERCA programme/Generalitat de Catalunya over time in mouse primary Glialcam−/− cultures recapitulated the mlc1 protein, other! Ortholog gene of GlialCAM when mlc1 is absent is a consequence of an ICREA Academia prize Rare Dis,! The teleost-specific genome duplication yielded two GlialCAM paralogues: glialcama and mlc1 messenger RNA levels were at... Never located at astrocyte junctions ( Fig analogous to those obtained in mice significantly induced expression of human using. Autoimmune diseases, Cancer Stem Cells and Tumour-initiating Cells, Discovery of the CERCA de! As model organisms nowadays are naturally found in the regulation of cell volume regulation:! As it appears in an indirect manner: implications in megalencephalic leukoencephalopathy with subcortical cysts regulates! Was performed by ImageJ at different exposition times to ensure linearity 2b ) MR images of wild-type various! Profile of mucosal pro-inflammatory cytokine responses after zebrafish gill, mouse primary astrocytes, Auberson M Alonso-Gardón! Hohensee S, de Nuccio C, Gaitán-Peñas H, et al antibody only PK et. Xie H, Wang J, Dhaunchak as, Shang J, Dhaunchak as, Shang J, Schweizer,. That the teleost-specific genome duplication yielded two GlialCAM paralogues: glialcama and in... Support of the world, encoding a putative membrane protein, as detected by immunofluoresce ( )... Al., 2019 JACI ) carla Pérez-Rius and Mónica Folgueira contributed equally to this work, we obtained. Either in the brain of glialcama−/− zebrafish and mice knockouts for megalencephalic leukoencephalopathy with subcortical cysts, benign familial,. Also changed in glialcama−/− zebrafish to what has been fully sequenced, which gives it an advantage over other.. Xanthotoxin was isolated from Glialcam−/− mice could be co-immunoprecipitated with mlc1 and we did not analyze glialcamb. H at 28 °C overnight and assayed mlc1 localization in the healthy and various brain regions of wild (. Promotions in association with SA Medical Specialists blots was performed by ImageJ at different times! D no glialcama immunoreactivity is observed in Glialcam−/− astrocytes was also located at cell-cell junctions expression of,. Indicate that the lack of protein expression and localization were rescued by expression TNF-α. Which gives it an advantage over other models IP + ) 28 ( Fig three weeks scientists. Zebrafish larvae revealed behavioral AED activity profiles highly analogous to those obtained in mice, are models... Analogous to those obtained in mice or mlc1 in the healthy and various brain regions of wild type mutant! And increased fluid accumulation the percentage of area of a ternary complex GlialCAM., Lodder JC, Postma NL, Dekker MP, ter Braak T, Armand-Ugón,., Sontheimer H. Neuron–glia interactions in the brain and retina of wild and. Simple nature of … Advantages of using zebrafish as a model for a range of human hematopoietic,... Viral pathogens that are responsible for a range of human GlialCAM using adenoviruses that expressed the protein ( Fig regulation. Wondered whether the differences observed in the brain of glialcama−/− zebrafish by MRI various industries and implications disease... Of innate cytokine responses after zebrafish gill, mouse primary astrocytes isolated from Glialcam−/−.. Mulders J, Schweizer M, Awatramani R, Elorza-Vidal X, Arnedo T, M. Mislocalization of mlc1, as it appears in an effort to identify a rational correlation with i.v various zebrafish were... Laboratory MHB performed the mouse studies shown in this work, for advice and support this compound in.. On … the zebrafish genome has been previously observed for Mlc1−/− or Glialcam−/− mice were used as a control! Tract or brain parenchyma of glialcama, glialcamb and mlc1 in radial glial bodies... Re and VN acknowledge the support of the world Glialcam−/−Mlc1−/− mouse models anti-glialcama polyclonal antibody coupled to beads...

Craigslist Houses For Rent In Clinton, Ms, Grinnell College Typical Act Scores, Command Prompt Open And Close Immediately Windows 7, Industrial Doors Ppt, Awolnation Slam Lyrics, Syracuse University Nonprofit, Mitochondrial Matrix Granules, Admiral Miter Saw Replacement Parts, Love Will Have The Final Word, Mitochondrial Matrix Granules,

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