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Keywords: Zebrafish
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Journal Articles
Dis Model Mech (2022) 15 (5): dmm049437.
Published: 9 May 2022
... in developing skeletal muscle remains unknown. To address this issue, we generated zebrafish spega , spegb and spega;spegb ( speg -DKO) mutant lines. We demonstrated that speg -DKO zebrafish faithfully recapitulate multiple phenotypes associated with CNM, including disruption of the ECC machinery, dysregulation...
Includes: Supplementary data
Journal Articles
Dis Model Mech dmm.044594.
Published: 6 May 2022
... of Hypermanganesemia with Dystonia 2, an inherited manganese transporter defect caused by mutations in SLC39A14. To identify novel potential targets of manganese neurotoxicity we performed transcriptome analysis of slc39a14 −/- mutant zebrafish unexposed and exposed to MnCl 2 . Differentially expressed genes mapped...
Journal Articles
Dis Model Mech dmm.049427.
Published: 28 April 2022
... assay in adult zebrafish using the bag3 gene knockout (bag3 e2/e2 ) cardiomyopathy model as a paradigm. First, utilizing a classic genetic breeding approach, we identified dnajb6b as a deleterious modifier gene for bag3 cardiomyopathy. Next, we established an F0-based genetic assay in adult zebrafish...
Journal Articles
Dis Model Mech (2022) 15 (4): dmm049109.
Published: 26 April 2022
... tissue samples and a long latency period for results. To overcome these issues, we established zebrafish GBM xenografts of diverse origin, which can tolerate intracranial engraftment and maintain their unique histological features. Subsequent single-cell RNA-sequencing (scRNA-seq) analysis confirmed...
Includes: Supplementary data
Journal Articles
Dis Model Mech (2022) 15 (4): dmm048921.
Published: 13 April 2022
.... However, the numerous copies in mammalian genomes and the lack of tools to study them make defining their role in health and diseases challenging. In this study, we identified eight copies of the zebrafish endogenous retrovirus zferv . We created and characterised the first in vivo ERV reporter line...
Includes: Supplementary data
Journal Articles
Dis Model Mech (2022) 15 (3): dmm049227.
Published: 29 March 2022
... medical therapies has not been successful. Zebrafish larvae offer a unique platform for drug screening to rapidly identify neuroprotective compounds following ICH. We applied the Spectrum Collection library compounds to zebrafish larvae acutely after ICH to screen for decreased brain cell death...
Includes: Supplementary data
Journal Articles
Dis Model Mech (2022) 15 (3): dmm046979.
Published: 28 March 2022
... growth factor beta (TGFβ) signaling play a causal or contributory role, respectively. Despite the advantages of cross-species disease modeling, animal models of aortic root aneurysm are largely restricted to genetically engineered mice. Here, we report that zebrafish devoid of the genes encoding latent...
Includes: Supplementary data
Journal Articles
Dis Model Mech (2022) 15 (3): dmm049094.
Published: 8 March 2022
... on improving symptoms rather than addressing the underlying causes of the disorder. Zebrafish offer many unique advantages for high-throughput preclinical evaluation of potential therapies for neurological diseases, including CDD. In particular, the large number of offspring produced, together...
Includes: Supplementary data
Journal Articles
In collection:
The RAS Pathway
Dis Model Mech (2022) 15 (2): dmm049099.
Published: 24 February 2022
... of protein mobility patterns inside cultured cells. In the present study, SMM was applied in vivo , using the zebrafish embryo model. We studied dynamics of the membrane protein H-Ras, its membrane-anchoring domain, C10H-Ras, and mutants, using total internal reflection fluorescence microscopy. Our results...
Includes: Supplementary data
Journal Articles
Dis Model Mech (2022) 15 (1): dmm049187.
Published: 26 January 2022
... from a collection of 11 early-onset familial Alzheimer's disease (EOFAD)-like and non-EOFAD-like mutations in three zebrafish genes. The one cellular activity consistently predicted as affected by only the EOFAD-like mutations is oxidative phosphorylation, which produces most of the energy of the brain...
Includes: Supplementary data
Journal Articles
Dis Model Mech (2021) 14 (12): dmm049145.
Published: 23 December 2021
... Mycobacterium tuberculosis isolates. In this study, we exploited the medium-throughput capabilities of the zebrafish embryo infection model with Mycobacterium marinum as a surrogate for M. tuberculosis . Using a representative set of clinically established drugs, we demonstrate that this model could...
Includes: Supplementary data
Journal Articles
Dis Model Mech (2021) 14 (12): dmm048965.
Published: 7 December 2021
... models only recapitulate auditory defects. We generated zebrafish mutants for one of the USH1 genes, protocadherin-15b ( pcdh15b ), a putative cell adhesion molecule. Zebrafish Pcdh15 is expressed exclusively in photoreceptors within calyceal processes (CPs), at the base of the outer segment (OS...
Includes: Supplementary data
Journal Articles
Dis Model Mech (2021) 14 (11): dmm049029.
Published: 11 November 2021
...) to promote vascular permeability. We show that genetic STAT3 ablation reduces vascular permeability in STAT3-deficient endothelium of mice and VEGF-inducible zebrafish crossed with CRISPR/Cas9-generated Stat3 knockout zebrafish. Intercellular adhesion molecule 1 (ICAM-1) expression is transcriptionally...
Includes: Supplementary data
Journal Articles
Dis Model Mech (2021) 14 (9): dmm049159.
Published: 16 September 2021
.... abscessus model of infection in CFTR-depleted zebrafish, associated with a striking increase in larval survival and reduction in pathological signs. The activity of Muddy was lost in macrophage-ablated larvae, suggesting that successful phage therapy relies on functional innate immunity. CFTR-depleted...
Includes: Supplementary data
Journal Articles
Dis Model Mech (2021) 14 (7): dmm047431.
Published: 23 July 2021
...Abdulsalam I. Isiaku; Zuobing Zhang; Vahid Pazhakh; Harriet R. Manley; Ella R. Thompson; Lucy C. Fox; Satwica Yerneni; Piers Blombery; Graham J. Lieschke ABSTRACT Zebrafish are an important model for studying phagocyte function, but rigorous experimental systems to distinguish whether phagocyte...
Includes: Supplementary data
Journal Articles
Dis Model Mech (2021) 14 (7): dmm048997.
Published: 23 July 2021
... subunits can develop sensorineural deafness, but the underlying mechanisms are unclear. Here, we show that V-ATPase mutations disrupt the formation of zebrafish neuromasts, which serve as a model to investigate hearing loss. V-ATPase mutant neuromasts are small and contain pyknotic nuclei that denote dying...
Includes: Supplementary data
Journal Articles
Dis Model Mech (2021) 14 (7): dmm049080.
Published: 7 July 2021
...Scott C. Baraban ABSTRACT Danio rerio (zebrafish) are a powerful experimental model for genetic and developmental studies. Adaptation of zebrafish to study seizures was initially established using the common convulsant agent pentylenetetrazole (PTZ). Larval PTZ-exposed zebrafish exhibit clear...
Journal Articles
Dis Model Mech (2021) 14 (5): dmm048439.
Published: 24 May 2021
... with cytoplasmic microtubules. Zebrafish ccdc103 / schmalhans ( smh ) mutants have macrophages and neutrophils with reduced proliferation, abnormally rounded cell morphology and an inability to migrate efficiently to the site of sterile wounds, all of which are consistent with a loss of cytoplasmic microtubule...
Includes: Supplementary data
Journal Articles
Dis Model Mech (2021) 14 (4): dmm047761.
Published: 26 April 2021
... disease spinal muscular atrophy (SMA). Compounds that can counteract synaptic destabilisation are rare. Here, we describe an automated screening paradigm in zebrafish for small-molecule compounds that stabilize the neuromuscular synapse in vivo . We make use of a mutant for the axonal C-type lectin...
Includes: Supplementary data
Journal Articles
Dis Model Mech (2020) 13 (11): dmm043885.
Published: 27 November 2020
...Alaa Koleilat; Joseph A. Dugdale; Trace A. Christenson; Jeffrey L. Bellah; Aaron M. Lambert; Mark A. Masino; Stephen C. Ekker; Lisa A. Schimmenti ABSTRACT The mariner ( myo7aa −/− ) mutant is a zebrafish model for Usher syndrome type 1 (USH1). To further characterize hair cell synaptic elements...
Includes: Supplementary data