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Modeling Developmental Processes and Disorders in Zebrafish
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Modeling Developmental Processes and Disorders in Zebrafish

A special issue of Cells (ISSN 2073-4409). This special issue belongs to the section "Cellular Biophysics".

Deadline for manuscript submissions: 31 December 2024 | Viewed by 18357

Special Issue Editors

Dr. Natascia Tiso

E-Mail Website
Guest Editor
Developmental Genetics Laboratory, Department of Biology, University of Padova, via Ugo Bassi 58/B, I-35131 Padova, Italy
Interests: zebrafish; heart disease; mitochondrial disease; endocrine disease; melanoma; neural development; pancreatic development and cancer
Special Issues, Collections and Topics in MDPI journals
Prof. Dr. Francesco Argenton

E-Mail Website
Guest Editor
Developmental Genetics Laboratory, Department of Biology, University of Padova, via Ugo Bassi 58/B, I-35131 Padova, Italy
Interests: zebrafish; cell fate decisions in pancreatic and neural development; genetic reporters for cellular studies

Special Issue Information

Dear Colleagues,

The Special Issue on “Modeling Developmental Processes and Disorders in Zebrafish” welcomes both reviews and original articles focusing on the application of the zebrafish (Danio rerio) organism to answer relevant biological questions on animal development and body functions under physiological or pathological conditions.

The Special Issue is inspired by the growing affirmation of the zebrafish vertebrate model in the field of Developmental Biology and Biomedical Sciences. The discovery of new processes of cell communication, differentiation, and organogenesis is inseparable from the progressive elucidation of the mechanisms underlying several human pathologies of still unknown etiology.

In this perspective, the zebrafish represents an ideal model for the in vivo study of both physiological and pathological processes given its transparency, high manipulability, affordability, and availability of a range of genetic tools, including mutant and transgenic lines, biosensors, and platforms for large-scale drug screening and proof-of-principle testing of innovative therapies.

Dr. Natascia Tiso
Prof. Dr. Francesco Argenton
Guest Editors

Manuscript Submission Information

Manuscripts should be submitted online at www.mdpi.com by registering and logging in to this website. Once you are registered, click here to go to the submission form. Manuscripts can be submitted until the deadline. All submissions that pass pre-check are peer-reviewed. Accepted papers will be published continuously in the journal (as soon as accepted) and will be listed together on the special issue website. Research articles, review articles as well as short communications are invited. For planned papers, a title and short abstract (about 100 words) can be sent to the Editorial Office for announcement on this website.

Submitted manuscripts should not have been published previously, nor be under consideration for publication elsewhere (except conference proceedings papers). All manuscripts are thoroughly refereed through a single-blind peer-review process. A guide for authors and other relevant information for submission of manuscripts is available on the Instructions for Authors page. Cells is an international peer-reviewed open access semimonthly journal published by MDPI.

Please visit the Instructions for Authors page before submitting a manuscript. The Article Processing Charge (APC) for publication in this open access journal is 2700 CHF (Swiss Francs). Submitted papers should be well formatted and use good English. Authors may use MDPI's English editing service prior to publication or during author revisions.

Published Papers (9 papers)

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Research

Jump to: Review

17 pages, 2306 KiB  
Article
Zebrafish CCNF and FUS Mediate Stress-Specific Motor Responses
by Yagiz Alp Aksoy, Alexander J Cole, Wei Deng and Daniel Hesselson
Cells 2024, 13(5), 372; https://doi.org/10.3390/cells13050372 - 21 Feb 2024
Viewed by 887
Abstract
Amyotrophic lateral sclerosis (ALS) is a devastating neurodegenerative disease characterized by the degeneration of motor neurons. Mutations in the cyclin F (CCNF) and fused in sarcoma (FUS) genes have been associated with ALS pathology. In this study, we aimed [...] Read more.
Amyotrophic lateral sclerosis (ALS) is a devastating neurodegenerative disease characterized by the degeneration of motor neurons. Mutations in the cyclin F (CCNF) and fused in sarcoma (FUS) genes have been associated with ALS pathology. In this study, we aimed to investigate the functional role of CCNF and FUS in ALS by using genome editing techniques to generate zebrafish models with genetic disruptions in these genes. Sequence comparisons showed significant homology between human and zebrafish CCNF and FUS proteins. We used CRISPR/Cas9 and TALEN-mediated genome editing to generate targeted disruptions in the zebrafish ccnf and fus genes. Ccnf-deficient zebrafish exhibited abnormal motor neuron development and axonal outgrowth, whereas Fus-deficient zebrafish did not exhibit developmental abnormalities or axonopathies in primary motor neurons. However, Fus-deficient zebrafish displayed motor impairments in response to oxidative and endoplasmic reticulum stress. The Ccnf-deficient zebrafish were only sensitized to endoplasmic reticulum stress, indicating that ALS genes have overlapping as well as unique cellular functions. These zebrafish models provide valuable platforms for studying the functional consequences of CCNF and FUS mutations in ALS pathogenesis. Furthermore, these zebrafish models expand the drug screening toolkit used to evaluate possible ALS treatments. Full article
(This article belongs to the Special Issue Modeling Developmental Processes and Disorders in Zebrafish)
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15 pages, 3303 KiB  
Article
Lymphatic Defects in Zebrafish sox18 Mutants Are Exacerbated by Perturbed VEGFC Signaling, While Masked by Elevated sox7 Expression
by Silvia Moleri, Sara Mercurio, Alex Pezzotta, Donatella D’Angelo, Alessia Brix, Alice Plebani, Giulia Lini, Marialaura Di Fuorti and Monica Beltrame
Cells 2023, 12(18), 2309; https://doi.org/10.3390/cells12182309 - 19 Sep 2023
Cited by 2 | Viewed by 1061
Abstract
Mutations in the transcription factor-coding gene SOX18, the growth factor-coding gene VEGFC and its receptor-coding gene VEGFR3/FLT4 cause primary lymphedema in humans. In mammals, SOX18, together with COUP-TFII/NR2F2, activates the expression of Prox1, a master regulator in lymphatic identity and development. [...] Read more.
Mutations in the transcription factor-coding gene SOX18, the growth factor-coding gene VEGFC and its receptor-coding gene VEGFR3/FLT4 cause primary lymphedema in humans. In mammals, SOX18, together with COUP-TFII/NR2F2, activates the expression of Prox1, a master regulator in lymphatic identity and development. Knockdown studies have also suggested an involvement of Sox18, Coup-tfII/Nr2f2, and Prox1 in zebrafish lymphatic development. Mutants in the corresponding genes initially failed to recapitulate the lymphatic defects observed in morphants. In this paper, we describe a novel zebrafish sox18 mutant allele, sa12315, which behaves as a null. The formation of the lymphatic thoracic duct is affected in sox18 homozygous mutants, but defects are milder in both zygotic and maternal-zygotic sox18 mutants than in sox18 morphants. Remarkably, in sox18 mutants, the expression of the closely related sox7 gene is elevated where lymphatic precursors arise. Sox7 could thus mask the absence of a functional Sox18 protein and account for the mild lymphatic phenotype in sox18 mutants, as shown in mice. Partial knockdown of vegfc exacerbates lymphatic defects in sox18 mutants, making them visible in heterozygotes. Our data thus reinforce the genetic interaction between Sox18 and Vegfc in lymphatic development, previously suggested by knockdown studies, and highlight the ability of Sox7 to compensate for Sox18 lymphatic dysfunction. Full article
(This article belongs to the Special Issue Modeling Developmental Processes and Disorders in Zebrafish)
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14 pages, 4694 KiB  
Article
Embryonic Ethanol but Not Cannabinoid Exposure Affects Zebrafish Cardiac Development via Agrin and Sonic Hedgehog Interaction
by Chengjin Zhang, Natalie Ezem, Shanta Mackinnon and Gregory J. Cole
Cells 2023, 12(9), 1327; https://doi.org/10.3390/cells12091327 - 06 May 2023
Viewed by 2037
Abstract
Recent studies demonstrate the adverse effects of cannabinoids on development, including via pathways shared with ethanol exposure. Our laboratory has shown that both the nervous system and cardiac development are dependent on agrin modulation of sonic hedgehog (shh) and fibroblast growth factor (Fgf) [...] Read more.
Recent studies demonstrate the adverse effects of cannabinoids on development, including via pathways shared with ethanol exposure. Our laboratory has shown that both the nervous system and cardiac development are dependent on agrin modulation of sonic hedgehog (shh) and fibroblast growth factor (Fgf) signaling pathways. As both ethanol and cannabinoids impact these signaling molecules, we examined their role on zebrafish heart development. Zebrafish embryos were exposed to a range of ethanol and/or cannabinoid receptor 1 and 2 agonist concentrations in the absence or presence of morpholino oligonucleotides that disrupt agrin or shh expression. In situ hybridization was employed to analyze cardiac marker gene expression. Exposure to cannabinoid receptor agonists disrupted midbrain–hindbrain boundary development, but had no effect on heart development, as assessed by the presence of cardiac edema or the altered expression of cardiac marker genes. In contrast, exposure to 1.5% ethanol induced cardiac edema and the altered expression of cardiac marker genes. Combined exposure to agrin or shh morpholino and 0.5% ethanol disrupted the cmlc2 gene expression pattern, with the restoration of the normal expression following shh mRNA overexpression. These studies provide evidence that signaling pathways critical to heart development are sensitive to ethanol exposure, but not cannabinoids, during early zebrafish embryogenesis. Full article
(This article belongs to the Special Issue Modeling Developmental Processes and Disorders in Zebrafish)
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13 pages, 31121 KiB  
Article
Development of a Triple-Negative Breast Cancer Leptomeningeal Disease Model in Zebrafish
by Udhayakumar Gopal, Jerry D. Monroe, Amarnath S. Marudamuthu, Salma Begum, Bradley J. Walters, Rodney A. Stewart, Chad W. Washington, Yann Gibert and Marcus A. Zachariah
Cells 2023, 12(7), 995; https://doi.org/10.3390/cells12070995 - 24 Mar 2023
Cited by 2 | Viewed by 2150
Abstract
Leptomeningeal disease occurs when cancer cells migrate into the ventricles of the brain and spinal cord and then colonize the meninges of the central nervous system. The triple-negative subtype of breast cancer often progresses toward leptomeningeal disease and has a poor prognosis because [...] Read more.
Leptomeningeal disease occurs when cancer cells migrate into the ventricles of the brain and spinal cord and then colonize the meninges of the central nervous system. The triple-negative subtype of breast cancer often progresses toward leptomeningeal disease and has a poor prognosis because of limited treatment options. This is due, in part, to a lack of animal models with which to study leptomeningeal disease. Here, we developed a translucent zebrafish casper (roy-/-; nacre-/-) xenograft model of leptomeningeal disease in which fluorescent labeled MDA-MB-231 human triple-negative breast cancer cells are microinjected into the ventricles of zebrafish embryos and then tracked and measured using fluorescent microscopy and multimodal plate reader technology. We then used these techniques to measure tumor area, cell proliferation, and cell death in samples treated with the breast cancer drug doxorubicin and a vehicle control. We monitored MDA-MB-231 cell localization and tumor area, and showed that samples treated with doxorubicin exhibited decreased tumor area and proliferation and increased apoptosis compared to control samples. Full article
(This article belongs to the Special Issue Modeling Developmental Processes and Disorders in Zebrafish)
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20 pages, 3041 KiB  
Article
Unraveling Presenilin 2 Functions in a Knockout Zebrafish Line to Shed Light into Alzheimer’s Disease Pathogenesis
by Lucia Barazzuol, Domenico Cieri, Nicola Facchinello, Tito Calì, Philip Washbourne, Francesco Argenton and Paola Pizzo
Cells 2023, 12(3), 376; https://doi.org/10.3390/cells12030376 - 19 Jan 2023
Cited by 2 | Viewed by 2333
Abstract
Mutations in presenilin 2 (PS2) have been causally linked to the development of inherited Alzheimer’s disease (AD). Besides its role as part of the γ-secretase complex, mammalian PS2 is also involved, as an individual protein, in a growing number of cell processes, which [...] Read more.
Mutations in presenilin 2 (PS2) have been causally linked to the development of inherited Alzheimer’s disease (AD). Besides its role as part of the γ-secretase complex, mammalian PS2 is also involved, as an individual protein, in a growing number of cell processes, which result altered in AD. To gain more insight into PS2 (dys)functions, we have generated a presenilin2 (psen2) knockout zebrafish line. We found that the absence of the protein does not markedly influence Notch signaling at early developmental stages, suggesting a Psen2 dispensable role in the γ-secretase-mediated Notch processing. Instead, loss of Psen2 induces an exaggerated locomotor response to stimulation in fish larvae, a reduced number of ER-mitochondria contacts in zebrafish neurons, and an increased basal autophagy. Moreover, the protein is involved in mitochondrial axonal transport, since its acute downregulation reduces in vivo organelle flux in zebrafish sensory neurons. Importantly, the expression of a human AD-linked mutant of the protein increases this vital process. Overall, our results confirm zebrafish as a good model organism for investigating PS2 functions in vivo, representing an alternative tool for the characterization of new AD-linked defective cell pathways and the testing of possible correcting drugs. Full article
(This article belongs to the Special Issue Modeling Developmental Processes and Disorders in Zebrafish)
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15 pages, 1529 KiB  
Article
Recapitulation of Retinal Damage in Zebrafish Larvae Infected with Zika Virus
by Adolfo Luis Almeida Maleski, Joao Gabriel Santos Rosa, Jefferson Thiago Gonçalves Bernardo, Renato Mancini Astray, Cristiani Isabel Banderó Walker, Monica Lopes-Ferreira and Carla Lima
Cells 2022, 11(9), 1457; https://doi.org/10.3390/cells11091457 - 26 Apr 2022
Cited by 5 | Viewed by 2152
Abstract
Zebrafish are increasingly being utilized as a model to investigate infectious diseases and to advance the understanding of pathogen–host interactions. Here, we take advantage of the zebrafish to recapitulate congenital ZIKV infection and, for the first time, demonstrate that it can be used [...] Read more.
Zebrafish are increasingly being utilized as a model to investigate infectious diseases and to advance the understanding of pathogen–host interactions. Here, we take advantage of the zebrafish to recapitulate congenital ZIKV infection and, for the first time, demonstrate that it can be used to model infection and reinfection and monitor anti-viral and inflammatory immune responses, as well as brain growth and eye abnormalities during embryonic development. By injecting a Brazilian strain of ZIKV into the yolk sac of one-cell stage embryos, we confirmed that, after 72 h, ZIKV successfully infected larvae, and the physical condition of the virus-infected hosts included gross morphological changes in surviving embryos (84%), with a reduction in larval head size and retinal damage characterized by increased thickness of the lens and inner nuclear layer. Changes in locomotor activity and the inability to perceive visual stimuli are a result of changes in retinal morphology caused by ZIKV. Furthermore, we demonstrated the ability of ZIKV to replicate in zebrafish larvae and infect new healthy larvae, impairing their visual and neurological functions. These data reinforce the deleterious activity of ZIKV in the brain and visual structures and establish the zebrafish as a model to study the molecular mechanisms involved in the pathology of the virus. Full article
(This article belongs to the Special Issue Modeling Developmental Processes and Disorders in Zebrafish)
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Review

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0 pages, 708 KiB  
Review
Utility of the Zebrafish Model for Studying Neuronal and Behavioral Disturbances Induced by Embryonic Exposure to Alcohol, Nicotine, and Cannabis
by Adam D. Collier, Abdul R. Abdulai and Sarah F. Leibowitz
Cells 2023, 12(20), 2505; https://doi.org/10.3390/cells12202505 - 23 Oct 2023
Cited by 1 | Viewed by 1396
Abstract
It is estimated that 5% of pregnant women consume drugs of abuse during pregnancy. Clinical research suggests that intake of drugs during pregnancy, such as alcohol, nicotine and cannabis, disturbs the development of neuronal systems in the offspring, in association with behavioral disturbances [...] Read more.
It is estimated that 5% of pregnant women consume drugs of abuse during pregnancy. Clinical research suggests that intake of drugs during pregnancy, such as alcohol, nicotine and cannabis, disturbs the development of neuronal systems in the offspring, in association with behavioral disturbances early in life and an increased risk of developing drug use disorders. After briefly summarizing evidence in rodents, this review focuses on the zebrafish model and its inherent advantages for studying the effects of embryonic exposure to drugs of abuse on behavioral and neuronal development, with an emphasis on neuropeptides known to promote drug-related behaviors. In addition to stimulating the expression and density of peptide neurons, as in rodents, zebrafish studies demonstrate that embryonic drug exposure has marked effects on the migration, morphology, projections, anatomical location, and peptide co-expression of these neurons. We also describe studies using advanced methodologies that can be applied in vivo in zebrafish: first, to demonstrate a causal relationship between the drug-induced neuronal and behavioral disturbances and second, to discover underlying molecular mechanisms that mediate these effects. The zebrafish model has great potential for providing important information regarding the development of novel and efficacious therapies for ameliorating the effects of early drug exposure. Full article
(This article belongs to the Special Issue Modeling Developmental Processes and Disorders in Zebrafish)
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34 pages, 1857 KiB  
Review
Zebrafish as a Useful Model System for Human Liver Disease
by Nobuyuki Shimizu, Hiroshi Shiraishi and Toshikatsu Hanada
Cells 2023, 12(18), 2246; https://doi.org/10.3390/cells12182246 - 11 Sep 2023
Cited by 2 | Viewed by 2218
Abstract
Liver diseases represent a significant global health challenge, thereby necessitating extensive research to understand their intricate complexities and to develop effective treatments. In this context, zebrafish (Danio rerio) have emerged as a valuable model organism for studying various aspects of liver [...] Read more.
Liver diseases represent a significant global health challenge, thereby necessitating extensive research to understand their intricate complexities and to develop effective treatments. In this context, zebrafish (Danio rerio) have emerged as a valuable model organism for studying various aspects of liver disease. The zebrafish liver has striking similarities to the human liver in terms of structure, function, and regenerative capacity. Researchers have successfully induced liver damage in zebrafish using chemical toxins, genetic manipulation, and other methods, thereby allowing the study of disease mechanisms and the progression of liver disease. Zebrafish embryos or larvae, with their transparency and rapid development, provide a unique opportunity for high-throughput drug screening and the identification of potential therapeutics. This review highlights how research on zebrafish has provided valuable insights into the pathological mechanisms of human liver disease. Full article
(This article belongs to the Special Issue Modeling Developmental Processes and Disorders in Zebrafish)
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31 pages, 1832 KiB  
Review
Bioelectricity in Developmental Patterning and Size Control: Evidence and Genetically Encoded Tools in the Zebrafish Model
by Martin R. Silic and GuangJun Zhang
Cells 2023, 12(8), 1148; https://doi.org/10.3390/cells12081148 - 13 Apr 2023
Cited by 4 | Viewed by 2792
Abstract
Developmental patterning is essential for regulating cellular events such as axial patterning, segmentation, tissue formation, and organ size determination during embryogenesis. Understanding the patterning mechanisms remains a central challenge and fundamental interest in developmental biology. Ion-channel-regulated bioelectric signals have emerged as a player [...] Read more.
Developmental patterning is essential for regulating cellular events such as axial patterning, segmentation, tissue formation, and organ size determination during embryogenesis. Understanding the patterning mechanisms remains a central challenge and fundamental interest in developmental biology. Ion-channel-regulated bioelectric signals have emerged as a player of the patterning mechanism, which may interact with morphogens. Evidence from multiple model organisms reveals the roles of bioelectricity in embryonic development, regeneration, and cancers. The Zebrafish model is the second most used vertebrate model, next to the mouse model. The zebrafish model has great potential for elucidating the functions of bioelectricity due to many advantages such as external development, transparent early embryogenesis, and tractable genetics. Here, we review genetic evidence from zebrafish mutants with fin-size and pigment changes related to ion channels and bioelectricity. In addition, we review the cell membrane voltage reporting and chemogenetic tools that have already been used or have great potential to be implemented in zebrafish models. Finally, new perspectives and opportunities for bioelectricity research with zebrafish are discussed. Full article
(This article belongs to the Special Issue Modeling Developmental Processes and Disorders in Zebrafish)
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Planned Papers

The below list represents only planned manuscripts. Some of these manuscripts have not been received by the Editorial Office yet. Papers submitted to MDPI journals are subject to peer-review.

Title: Diving Deep: Zebrafish Model in Motor Neuron Degeneration Research
Authors: Vranda Garg; Bart R.H. Geurten
Affiliation: Georg-August-University Göttingen, Department of Cellular Neurobiology, Julia-Lermontowa-Weg 3 37077 Göttingen, Germany; University of Otago, Department of Zoology, 340 Great King Street, 9016 Dunedin, New Zealand
Abstract: In the ever-evolving landscape of biomedical science, the pursuit of effective treatments for motor neuron disorders like amyotrophic lateral sclerosis (ALS), hereditary spastic paraplegia (HSP), spinal muscular atrophy (SMA) is a key priority. A crucial aspect of this endeavour is the development of robust animal models, and the zebrafish stands out as a prime candidate. This model offers significant research potential with its embryonic transparency, rapid life cycle, and notable genetic and neuroanatomical similarities with humans. Despite the difference in locomotion - zebrafish undulate while humans use limbs, the zebrafish presents relevant phenotypic parallels to human motor control disorders, providing valuable insights into neurodegenerative diseases. This review delves into how the zebrafish, through its inherent traits, offers significant insights into the complex behavioural and cellular phenotypes associated with these disorders. Furthermore, we examine recent advancements in high-throughput drug screening using the zebrafish model, a promising avenue for identifying therapeutically potent compounds.

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