Zebrafish models of inherited retinal dystrophies

Perkins, Brian D.

doi:10.20517/jtgg.2021.47

Cited by 7 publications

(9 citation statements)

References 155 publications

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“…Metabonomics usually regards biology as a whole and focuses on exploring the relationship between internal organs, tissues, and endogenous substances. At the same time, it can also show a series of biological events that occur in the physiological activities of its body under the influence of external and internal factors [ 10 ]. Since the 1980s, metabonomics has begun to sprout.…”

Section: Literature Reviewmentioning

confidence: 99%

[Retracted] Analysis of Metabonomic Characteristics after Exercise Fatigue Based on NMR

Chen

Wang

2022

Contrast Media & Molecular Imaging

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In order to better understand the changes of body metabolism of athletes before and after exercise, this paper proposes a method of metabonomics feature analysis after exercise fatigue based on NMR. This paper makes a retrospective analysis on the metabonomic characteristics of urine of middle and long distance runners after 30 minutes of a heavy load training class based on NMR. The experiment analyzes the urine of athletes by combining NMR technology and metabonomics principle. The experimental results show that the Q2 value of this experiment is greater than that of all comparisons, and r2x and r2y are 0.436 and 0.85, respectively. Conclusion. This method can effectively analyze the changes of body metabolism after athletes’ fatigue, so as to better alleviate athletes’ fatigue after exercise.

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Section: Literature Reviewmentioning

confidence: 99%

[Retracted] Analysis of Metabonomic Characteristics after Exercise Fatigue Based on NMR

Chen

Wang

2022

Contrast Media & Molecular Imaging

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“…Conversely, retinal degenerative diseases typically involve thez slow progressive loss of specific cell types, such as rod photoreceptors in retinitis pigmentosa 61 . Interestingly, several zebrafish mutants fail to regenerate retinal neurons when loss is slow, progressive, and cell-type specific 46 , i.e., akin to disease condtions 46 . This suggests that a threshold of cell death is required to trigger retinal regeneration and that models enabling control over the onset, extent, specificity and duration of cell death can expand our understanding of how regeneration is controlled under disease-mimicking conditions.…”

Section: Discussionmentioning

confidence: 99%

“…While several mutant zebrafish lines exhibit cell-type specific losses, the lack of control over the onset and duration of cell death make it more difficult to profile regenerative responses in these models. Additionally, several retinal degeneration mutants also fail to regenerate 44 suggesting that a threshold of cell death is required to trigger retinal regeneration. Third, co-expression of a fluorescent reporter in NTR-targeted cells allows the regenerative process to be characterized in detail using in vivo time-lapse imaging 38,45 or quantified using high-throughput methods 39,46 .…”

Section: Introductionmentioning

confidence: 99%

“…While several mutant zebrafish lines exhibit cell-type specific losses, the lack of control over the onset and duration of cell death make it more difficult to profile regenerative responses in these models. Additionally, several retinal degeneration mutants also fail to regenerate 46 suggesting that a threshold of cell death is required to trigger retinal regeneration. Using this approach, recent studies have shown that selective retinal cell loss elicits a fate-biased regenerative response [13][14][15][16] , although subtler fate-biases have also recently been acknowledged in widespread injury paradigms as well.…”

Section: Introductionmentioning

confidence: 99%

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Transcriptomic comparison of two selective retinal cell ablation paradigms in zebrafish reveals shared and cell-specific regenerative responses

Walker

Wang

Emmerich

et al. 2020

Preprint

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Zebrafish are an effective model organism for retinal regeneration studies. Regenerated retinal cells are derived from Müller glia (MG) stem cells. Mammalian MG can also produce new retinal neurons; however, this regenerative potential remains dormant in the absence of genetic and/or chemical stimulation. An understanding of how the regenerative potential of MG is regulated could aid efforts to promote regeneration therapeutically. Following widespread retinal cell death, developmental signaling coordinates regeneration. Less is known about how MG respond to the loss of specific cell types, i.e., paradigms similar to retinal degenerative diseases. To address this, transcriptomic responses to the selective loss of rod photoreceptors or retinal bipolar cells were compared over twelve timepoints spanning cell degeneration and regeneration. Shared and paradigm-specific expression changes were identified throughout regeneration. Overall, paradigm-specific changes predominated, suggesting cell-specific mechanisms for activating MG stem cells. One particularly interested finding new for retinal regeneration was early regulation of SOCS family genes. These are associated with stat3 activation and the JAK/STAT pathway which has been well documented in similar studies and was further supported in our analyses. These data support the concept that selective retinal cell loss can elicit cell-specific regenerative programs and provide novel insights into retinal regeneration.

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“…Overall, the amount of genetic information available from zebrafish mutants, including evolutionary well-conserved factors associated with the possibility of inducing visual impairment, supports zebrafish as a powerful model for understanding visual disorders. However, besides the exceptional ability of the zebrafish retina to regenerate after injury, MG failed to proliferate and regenerate damaged cells in several zebrafish models: for instance, mutants bbs2 (BBS disorders), MZcep290fb208 (retinal dystrophy), cerkl (nonsyndromic RP and cone-rod dystrophy), eys (autosomal recessive RP), rp2 (X-linked RP) rpgrip1 (RP GTPase regulator interacting protein 1, X-linked RP) [ 30 ]. In rats and mice, extensive effort is paid to manipulate MG to induce their regenerative ability and/or reprogramming potential, especially for substitute photoreceptors and RGCs, which are the most affected populations of the mammalian retina during degenerative diseases that cause blindness.…”

Section: Alternative Organism Models For Retina Neuroregenerationmentioning

confidence: 99%

Regenerative Strategies for Retinal Neurons: Novel Insights in Non-Mammalian Model Organisms

Catalani

Cherubini

Quondam

et al. 2022

IJMS

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A detailed knowledge of the status of the retina in neurodegenerative conditions is a crucial point for the development of therapeutics in retinal pathologies and to translate eye research to CNS disease. In this context, manipulating signaling pathways that lead to neuronal regeneration offers an excellent opportunity to substitute damaged cells and, thus, restore the tissue functionality. Alternative systems and methods are increasingly being considered to replace/reduce in vivo approaches in the study of retina pathophysiology. Herein, we present recent data obtained from the zebrafish (Danio rerio) and the fruit fly Drosophila melanogaster that bring promising advantages into studying and modeling, at a preclinical level, neurodegeneration and regenerative approaches in retinal diseases. Indeed, the regenerative ability of vertebrate model zebrafish is particularly appealing. In addition, the fruit fly is ideal for regenerative studies due to its high degree of conservation with vertebrates and the broad spectrum of genetic variants achievable. Furthermore, a large part of the drosophila brain is dedicated to sight, thus offering the possibility of studying common mechanisms of the visual system and the brain at once. The knowledge acquired from these alternative models may help to investigate specific well-conserved factors of interest in human neuroregeneration after injuries or during pathologies.

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Zebrafish models of inherited retinal dystrophies

Cited by 7 publications

References 155 publications

[Retracted] Analysis of Metabonomic Characteristics after Exercise Fatigue Based on NMR

[Retracted] Analysis of Metabonomic Characteristics after Exercise Fatigue Based on NMR

Transcriptomic comparison of two selective retinal cell ablation paradigms in zebrafish reveals shared and cell-specific regenerative responses

Regenerative Strategies for Retinal Neurons: Novel Insights in Non-Mammalian Model Organisms

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