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Keywords: Zebrafish
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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 dmm.049099.
Published: 20 December 2021
... 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 (TIRFM). Our results...
Journal Articles
Dis Model Mech dmm.049094.
Published: 16 December 2021
... on improving symptoms rather than addressing the underlying causes of the disorder. Zebrafish offer many unique advantages for high-throughput pre-clinical evaluation of potential therapies for neurological diseases, including CDD. In particular, the large number of offspring produced, together...
Journal Articles
Dis Model Mech (2021) 14 (12): dmm048965.
Published: 07 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 dmm.049187.
Published: 29 November 2021
... a collection of eleven 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 that produces most of the brain's energy. All...
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: 07 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
Journal Articles
Dis Model Mech (2020) 13 (9): dmm045971.
Published: 24 September 2020
...Jelmer Hoeksma; Gerard C. M. van der Zon; Peter ten Dijke; Jeroen den Hertog ABSTRACT Zebrafish models are well-established tools for investigating the underlying mechanisms of diseases. Here, we identified cercosporamide, a metabolite from the fungus Ascochyta aquiliqiae , as a potent bone...
Includes: Supplementary data
Journal Articles
Dis Model Mech (2020) 13 (8): dmm045716.
Published: 23 August 2020
.... The preclinical evaluation of novel tuberculosis vaccine candidates is also hampered by the lack of an appropriate animal model that could accurately predict the protective effect of vaccines in humans. Here, we review the role of zebrafish ( Danio rerio ) and other fish models in the development of novel...
Journal Articles
Dis Model Mech (2020) 13 (7): dmm043091.
Published: 20 July 2020
... immune responses observed in mouse versus human cells. Here, we developed a zebrafish infection model to study the innate immune response to Toxoplasma in vivo . By infecting the zebrafish hindbrain ventricle, and using high-resolution microscopy techniques coupled with computer vision-driven automated...
Includes: Supplementary data
Journal Articles
Dis Model Mech (2020) 13 (6): dmm042556.
Published: 26 June 2020
... fluorescent reporter in a dose-dependent manner. Furthermore, cytokine misexpression is enabled only in tissue domains that can be defined by Cre recombinase expression. We have validated this system in zebrafish using an insulin : cre line. In doubly transgenic fish, quantitative real-time polymerase chain...
Includes: Supplementary data
Journal Articles
Dis Model Mech (2020) 13 (6): dmm042549.
Published: 24 June 2020
... control of bone development and provide new zebrafish models for human disorders of the cholesterol biosynthesis pathway. Here, for the first time, we show that msmo1 and lss are needed for proper chondrocyte differentiation, growth plate formation and endochondral ossification. To our knowledge...
Includes: Supplementary data
Journal Articles
Dis Model Mech (2020) 13 (5): dmm044131.
Published: 27 May 2020
... to regenerate after injury. In stark contrast, the regeneration of a completely transected spinal cord and accompanying reversal of paralysis in adult zebrafish is arguably one of the most spectacular biological phenomena in nature. Here, we review reports from the last decade that dissect the mechanisms...
Journal Articles
Dis Model Mech (2020) 13 (4): dmm043877.
Published: 29 April 2020
... phenotypes have been generated, the underlying molecular mechanisms remain largely elusive. Over the past decades, the zebrafish has emerged as a powerful vertebrate model that has led to fundamental discoveries in developmental neurobiology and behavioral genetics. Here, we review the attributes that make...