Search for Articles:
Title / Keyword
Author / Affiliation / Email
Journal
Article Type
 
 
Advanced Search
 
Section
Special Issue
Volume
Issue
Number
Page
 
7.8
5.6
Journals
IJMS
Special Issues
Zebrafish as a Model in Human Disease 2.0
ijms-logo
Submit to IJMS Review for IJMS Propose a Special Issue

Journal Menu

► Journal Menu

Journal Browser

► Journal Browser
share announcement

Zebrafish as a Model in Human Disease 2.0

A special issue of International Journal of Molecular Sciences (ISSN 1422-0067). This special issue belongs to the section "Molecular Pathology, Diagnostics, and Therapeutics".

Deadline for manuscript submissions: closed (28 December 2023) | Viewed by 18551

Special Issue Editors

Dr. Marco G. Schiavone

E-Mail Website
Guest Editor
Department of Molecular and Translational Medicine, Zebrafish Facility, University of Brescia, 25123 Brescia, Italy
Interests: zebrafish; Ca2+ signalling; molecular pathways; mitochondria; muscular dystrophy; CRISPR/Cas9; transgenic reporter lines; RNAseq; biosensors; generation of zebrafish models for human disease; drug screening and development; OCR measurement
Special Issues, Collections and Topics in MDPI journals
Dr. Nicola Facchinello

E-Mail Website
Guest Editor
Neuroscience Institute, National Research Council (CNR), 35131 Padua, Italy
Interests: zebrafish; CRISPR/Cas9; transgenic lines; biosensors; endothelial cells; extracellular matrix; genetic mutant for human disease modelling; xenografts
Special Issues, Collections and Topics in MDPI journals
Dr. Andrea Vettori

E-Mail Website
Guest Editor
Department of Biotechnology, University of Verona, 37134 Verona, Italy
Interests: molecular genetics; arrhythmogenic cardiomyopathy; hereditary cancers; epilepsy; Hereditary Spastic Paraplegia (HSP); zebrafish mutant models
Special Issues, Collections and Topics in MDPI journals

Special Issue Information

Dear Colleagues,

This Special Issue is the continuation of our previous Special Issue, “Zebrafish as a Model in Human Disease”. We welcome original investigations as well as concise review manuscripts focusing on the application of the zebrafish organism to model disease and provide new insight into disease mechanisms and therapy. Understanding and fighting diseases require the right tools. Some aspects of disease biology and treatment, such as tissue homeostasis, interactions between cells and their microenvironment, and response to drugs, cannot be fully captured in vitro. The zebrafish continues to fascinate the research community and represents an ideal model for the in vivo study given its transparency, high manipulability, versatility and availability of a range of genetic tools (mutant, transgenic lines and biosensors), enabling researchers to answer key biological questions at a whole-organism level. In addition to classical approaches, recent technological advances for zebrafish disease modeling have been in precision genome editing. CRISPR-Cas9 has enabled the efficient generation of genetic mutations in zebrafish, and in particular, this can be used to generate site-specific or tissue-specific mutations. The current successes in this area make it even more exciting to look forward to new disease models and tools to investigate them, resulting in more robust, representative and predictive preclinical models.

This Special Issue seeks to attract either basic studies of human genetic diseases in zebrafish, as well as studies describing new methods and tools for genome editing to investigate these models and studies for large-scale drug screening that will guide new avenues for therapeutics development.

Dr. Marco G. Schiavone
Dr. Nicola Facchinello
Dr. Andrea Vettori
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. International Journal of Molecular Sciences is an international peer-reviewed open access semimonthly journal published by MDPI.

Please visit the Instructions for Authors page before submitting a manuscript. There is an Article Processing Charge (APC) for publication in this open access journal. For details about the APC please see here. 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.

 

Keywords

  • zebrafish
  • CRISPR/Cas9
  • transgenic lines
  • biosensors
  • endothelial cells
  • extracellular matrix
  • genetic mutant for human disease modeling
  • xenografts

Published Papers (11 papers)

Download All Papers
Order results
Result details
Show export options expand_more Show export options expand_less
Select all
Export citation of selected articles as:

Research

Jump to: Review

25 pages, 4499 KiB  
Article
A Zebrafish-Based Platform for High-Throughput Epilepsy Modeling and Drug Screening in F0
by Sílvia Locubiche, Víctor Ordóñez, Elena Abad, Michele Scotto di Mase, Vincenzo Di Donato and Flavia De Santis
Int. J. Mol. Sci. 2024, 25(5), 2991; https://doi.org/10.3390/ijms25052991 - 4 Mar 2024
Viewed by 1166
Abstract
The zebrafish model has emerged as a reference tool for phenotypic drug screening. An increasing number of molecules have been brought from bench to bedside thanks to zebrafish-based assays over the last decade. The high homology between the zebrafish and the human genomes [...] Read more.
The zebrafish model has emerged as a reference tool for phenotypic drug screening. An increasing number of molecules have been brought from bench to bedside thanks to zebrafish-based assays over the last decade. The high homology between the zebrafish and the human genomes facilitates the generation of zebrafish lines carrying loss-of-function mutations in disease-relevant genes; nonetheless, even using this alternative model, the establishment of isogenic mutant lines requires a long generation time and an elevated number of animals. In this study, we developed a zebrafish-based high-throughput platform for the generation of F0 knock-out (KO) models and the screening of neuroactive compounds. We show that the simultaneous inactivation of a reporter gene (tyrosinase) and a second gene of interest allows the phenotypic selection of F0 somatic mutants (crispants) carrying the highest rates of mutations in both loci. As a proof of principle, we targeted genes associated with neurodevelopmental disorders and we efficiently generated de facto F0 mutants in seven genes involved in childhood epilepsy. We employed a high-throughput multiparametric behavioral analysis to characterize the response of these KO models to an epileptogenic stimulus, making it possible to employ kinematic parameters to identify seizure-like events. The combination of these co-injection, screening and phenotyping methods allowed us to generate crispants recapitulating epilepsy features and to test the efficacy of compounds already during the first days post fertilization. Since the strategy can be applied to a wide range of indications, this study paves the ground for high-throughput drug discovery and promotes the use of zebrafish in personalized medicine and neurotoxicity assessment. Full article
(This article belongs to the Special Issue Zebrafish as a Model in Human Disease 2.0)
►▼ Show Figures

Figure 1

16 pages, 4687 KiB  
Article
Dynamic Regulation of brsk2 in the Social and Motor Development of Zebrafish: A Developmental Behavior Analysis
by Jingxin Deng, Chunxue Liu, Meixin Hu, Chunchun Hu, Jia Lin, Qiang Li and Xiu Xu
Int. J. Mol. Sci. 2023, 24(22), 16506; https://doi.org/10.3390/ijms242216506 - 19 Nov 2023
Cited by 1 | Viewed by 1064
Abstract
Both social and motor development play an essential role in an individual’s physical, psychological, and social well-being. It is essential to conduct a dynamic analysis at multiple time points during the developmental process as it helps us better understand and evaluate the trajectory [...] Read more.
Both social and motor development play an essential role in an individual’s physical, psychological, and social well-being. It is essential to conduct a dynamic analysis at multiple time points during the developmental process as it helps us better understand and evaluate the trajectory and changes in individual development. Recently, some studies found that mutations in the BRSK2 gene may contribute to motor impairments, delays in achieving motor milestones, and deficits in social behavior and communication skills in patients. However, little is known about the dynamic analysis of social and motor development at multiple time points during the development of the brsk2 gene. We generated a novel brsk2-deficient (brsk2ab−/−) zebrafish model through CRISPR/Cas9 editing and conducted comprehensive morphological and neurobehavioral evaluations, including that of locomotor behaviors, social behaviors, and anxiety behaviors from the larval to adult stages of development. Compared to wild-type zebrafish, brsk2ab−/− zebrafish exhibited a catch-up growth pattern of body length and gradually improved locomotor activities during the developmental process. In contrast, multimodal behavior tests showed that the brsk2ab−/− zebrafish displayed escalating social deficiency and anxiety-like behaviors over time. We reported for the first time that the brsk2 gene had dynamic regulatory effects on motor and social development. It helps us understand developmental trends, capture changes, facilitate early interventions, and provide personalized support and development opportunities for individuals. Full article
(This article belongs to the Special Issue Zebrafish as a Model in Human Disease 2.0)
►▼ Show Figures

Figure 1

23 pages, 2209 KiB  
Article
The Neurotoxic Effect of Environmental Temperature Variation in Adult Zebrafish (Danio rerio)
by Elisa Maffioli, Simona Nonnis, Francesca Grassi Scalvini, Armando Negri, Gabriella Tedeschi and Mattia Toni
Int. J. Mol. Sci. 2023, 24(21), 15735; https://doi.org/10.3390/ijms242115735 - 29 Oct 2023
Cited by 1 | Viewed by 1178
Abstract
Neurotoxicity consists of the altered functionality of the nervous system caused by exposure to chemical agents or altered chemical–physical parameters. The neurotoxic effect can be evaluated from the molecular to the behavioural level. The zebrafish Danio rerio is a model organism used in [...] Read more.
Neurotoxicity consists of the altered functionality of the nervous system caused by exposure to chemical agents or altered chemical–physical parameters. The neurotoxic effect can be evaluated from the molecular to the behavioural level. The zebrafish Danio rerio is a model organism used in many research fields, including ecotoxicology and neurotoxicology. Recent studies by our research group have demonstrated that the exposure of adult zebrafish to low (18 °C) or high (34 °C) temperatures alters their brain proteome and fish behaviour compared to control (26 °C). These results showed that thermal variation alters the functionality of the nervous system, suggesting a temperature-induced neurotoxic effect. To demonstrate that temperature variation can be counted among the factors that generate neurotoxicity, eight different protein datasets, previously published by our research group, were subjected to new analyses using an integrated proteomic approach by means of the Ingenuity Pathway Analysis (IPA) software (Release December 2022). The datasets consist of brain proteome analyses of wild type adult zebrafish kept at three different temperatures (18 °C, 26 °C, and 34 °C) for 4 days (acute) or 21 days (chronic treatment), and of BDNF+/− and BDNF−/− zebrafish kept at 26 °C or 34 °C for 21 days. The results (a) demonstrate that thermal alterations generate an effect that can be defined as neurotoxic (p value ≤ 0.05, activation Z score ≤ −2 or ≥2), (b) identify 16 proteins that can be used as hallmarks of the neurotoxic processes common to all the treatments applied and (c) provide three protein panels (p value ≤ 0.05) related to 18 °C, 34 °C, and BDNF depletion that can be linked to anxiety-like or boldness behaviour upon these treatments. Full article
(This article belongs to the Special Issue Zebrafish as a Model in Human Disease 2.0)
►▼ Show Figures

Figure 1

18 pages, 3470 KiB  
Article
Downregulation of Zebrafish Cytosolic Sialidase Neu3.2 Affects Skeletal Muscle Development
by Daniela Zizioli, Silvia Codenotti, Giuliana Benaglia, Marta Manzoni, Elena Massardi, Alessandro Fanzani, Giuseppe Borsani and Eugenio Monti
Int. J. Mol. Sci. 2023, 24(17), 13578; https://doi.org/10.3390/ijms241713578 - 1 Sep 2023
Viewed by 1105
Abstract
Sialidases remove terminal sialic acids residues from the non-reducing ends of glycoconjugates. They have been recognized as catabolic enzymes that work within different subcellular compartments and can ensure the proper turn-over of glycoconjugates. Four mammalian sialidases (NEU1-4) exist, with different subcellular localization, pH [...] Read more.
Sialidases remove terminal sialic acids residues from the non-reducing ends of glycoconjugates. They have been recognized as catabolic enzymes that work within different subcellular compartments and can ensure the proper turn-over of glycoconjugates. Four mammalian sialidases (NEU1-4) exist, with different subcellular localization, pH optimum and substrate specificity. In zebrafish, seven different sialidases, with high homology to mammalian counterparts, have been identified. Zebrafish Neu3.2 is similar to the human cytosolic sialidase NEU2, which is involved in skeletal muscle differentiation and exhibits a broad substrate specificity toward gangliosides and glycoproteins. In zebrafish neu3.2, mRNA is expressed during somite development, and its enzymatic activity has been detected in the skeletal muscle and heart of adult animals. In this paper, 1–4-cell-stage embryos injected with neu3.2 splice-blocking morpholino showed severe embryonic defects, mainly in somites, heart and anterior–posterior axis formation. Myog and myod1 expressions were altered in morphants, and impaired musculature formation was associated with a defective locomotor behavior. Finally, the co-injection of Neu2 mouse mRNA in morphants rescued the phenotype. These data are consistent with the involvement of cytosolic sialidase in pathologies related to muscle formation and support the validity of the model to investigate the pathogenesis of the diseases. Full article
(This article belongs to the Special Issue Zebrafish as a Model in Human Disease 2.0)
►▼ Show Figures

Figure 1

24 pages, 4465 KiB  
Article
Modeling Sarcoglycanopathy in Danio rerio
by Francesco Dalla Barba, Michela Soardi, Leila Mouhib, Giovanni Risato, Eylem Emek Akyürek, Tyrone Lucon-Xiccato, Martina Scano, Alberto Benetollo, Roberta Sacchetto, Isabelle Richard, Francesco Argenton, Cristiano Bertolucci, Marcello Carotti and Dorianna Sandonà
Int. J. Mol. Sci. 2023, 24(16), 12707; https://doi.org/10.3390/ijms241612707 - 11 Aug 2023
Viewed by 1372
Abstract
Sarcoglycanopathies, also known as limb girdle muscular dystrophy 3-6, are rare muscular dystrophies characterized, although heterogeneous, by high disability, with patients often wheelchair-bound by late adolescence and frequently developing respiratory and cardiac problems. These diseases are currently incurable, emphasizing the importance of effective [...] Read more.
Sarcoglycanopathies, also known as limb girdle muscular dystrophy 3-6, are rare muscular dystrophies characterized, although heterogeneous, by high disability, with patients often wheelchair-bound by late adolescence and frequently developing respiratory and cardiac problems. These diseases are currently incurable, emphasizing the importance of effective treatment strategies and the necessity of animal models for drug screening and therapeutic verification. Using the CRISPR/Cas9 genome editing technique, we generated and characterized δ-sarcoglycan and β-sarcoglycan knockout zebrafish lines, which presented a progressive disease phenotype that worsened from a mild larval stage to distinct myopathic features in adulthood. By subjecting the knockout larvae to a viscous swimming medium, we were able to anticipate disease onset. The δ-SG knockout line was further exploited to demonstrate that a δ-SG missense mutant is a substrate for endoplasmic reticulum-associated degradation (ERAD), indicating premature degradation due to protein folding defects. In conclusion, our study underscores the utility of zebrafish in modeling sarcoglycanopathies through either gene knockout or future knock-in techniques. These novel zebrafish lines will not only enhance our understanding of the disease’s pathogenic mechanisms, but will also serve as powerful tools for phenotype-based drug screening, ultimately contributing to the development of a cure for sarcoglycanopathies. Full article
(This article belongs to the Special Issue Zebrafish as a Model in Human Disease 2.0)
►▼ Show Figures

Figure 1

18 pages, 3449 KiB  
Article
The ndrg2 Gene Regulates Hair Cell Morphogenesis and Auditory Function during Zebrafish Development
by Cheng Wang, Xin Wang, Hao Zheng, Jia Yao, Yuqing Xiang and Dong Liu
Int. J. Mol. Sci. 2023, 24(12), 10002; https://doi.org/10.3390/ijms241210002 - 11 Jun 2023
Viewed by 1493
Abstract
Damages of sensory hair cells (HCs) are mainly responsible for sensorineural hearing loss, however, its pathological mechanism is not yet fully understood due to the fact that many potential deafness genes remain unidentified. N-myc downstream-regulated gene 2 (ndrg2) is commonly regarded [...] Read more.
Damages of sensory hair cells (HCs) are mainly responsible for sensorineural hearing loss, however, its pathological mechanism is not yet fully understood due to the fact that many potential deafness genes remain unidentified. N-myc downstream-regulated gene 2 (ndrg2) is commonly regarded as a tumor suppressor and a cell stress-responsive gene extensively involved in cell proliferation, differentiation, apoptosis and invasion, while its roles in zebrafish HC morphogenesis and hearing remains unclear. Results of this study suggested that ndrg2 was highly expressed in the HCs of the otic vesicle and neuromasts via in situ hybridization and single-cell RNA sequencing. Ndrg2 loss-of-function larvae showed decreased crista HCs, shortened cilia, and reduced neuromasts and functional HCs, which could be rescued by the microinjection of ndrg2 mRNA. Moreover, ndrg2 deficiency induced attenuated startle response behaviors to sound vibration stimuli. Mechanistically, there were no detectable HC apoptosis and supporting cell changes in the ndrg2 mutants, and HCs were capable of recovering by blocking the Notch signaling pathway, suggesting that ndrg2 was implicated in HC differentiation mediated by Notch. Overall, our study demonstrates that ndrg2 plays crucial roles in HC development and auditory sensory function utilizing the zebrafish model, which provides new insights into the identification of potential deafness genes and regulation mechanism of HC development. Full article
(This article belongs to the Special Issue Zebrafish as a Model in Human Disease 2.0)
►▼ Show Figures

Figure 1

20 pages, 2863 KiB  
Article
Deficiency of AP1 Complex Ap1g1 in Zebrafish Model Led to Perturbation of Neurodevelopment, Female and Male Fertility; New Insight to Understand Adaptinopathies
by Luca Mignani, Nicola Facchinello, Marco Varinelli, Elena Massardi, Natascia Tiso, Cosetta Ravelli, Stefania Mitola, Peter Schu, Eugenio Monti, Dario Finazzi, Giuseppe Borsani and Daniela Zizioli
Int. J. Mol. Sci. 2023, 24(8), 7108; https://doi.org/10.3390/ijms24087108 - 12 Apr 2023
Cited by 1 | Viewed by 1608
Abstract
In vertebrates, two homologous heterotetrameric AP1 complexes regulate the intracellular protein sorting via vesicles. AP-1 complexes are ubiquitously expressed and are composed of four different subunits: γ, β1, μ1 and σ1. Two different complexes are present in eukaryotic cells, AP1G1 (contains γ1 subunit) [...] Read more.
In vertebrates, two homologous heterotetrameric AP1 complexes regulate the intracellular protein sorting via vesicles. AP-1 complexes are ubiquitously expressed and are composed of four different subunits: γ, β1, μ1 and σ1. Two different complexes are present in eukaryotic cells, AP1G1 (contains γ1 subunit) and AP1G2 (contains γ2 subunit); both are indispensable for development. One additional tissue-specific isoform exists for μ1A, the polarized epithelial cells specific to μ1B; two additional tissue-specific isoforms exist for σ1A: σ1B and σ1C. Both AP1 complexes fulfil specific functions at the trans-Golgi network and endosomes. The use of different animal models demonstrated their crucial role in the development of multicellular organisms and the specification of neuronal and epithelial cells. Ap1g1 (γ1) knockout mice cease development at the blastocyst stage, while Ap1m1 (μ1A) knockouts cease during mid-organogenesis. A growing number of human diseases have been associated with mutations in genes encoding for the subunits of adaptor protein complexes. Recently, a new class of neurocutaneous and neurometabolic disorders affecting intracellular vesicular traffic have been referred to as adaptinopathies. To better understand the functional role of AP1G1 in adaptinopathies, we generated a zebrafish ap1g1 knockout using CRISPR/Cas9 genome editing. Zebrafish ap1g1 knockout embryos cease their development at the blastula stage. Interestingly, heterozygous females and males have reduced fertility and showed morphological alterations in the brain, gonads and intestinal epithelium. An analysis of mRNA profiles of different marker proteins and altered tissue morphologies revealed dysregulated cadherin-mediated cell adhesion. These data demonstrate that the zebrafish model organism enables us to study the molecular details of adaptinopathies and thus also develop treatment strategies. Full article
(This article belongs to the Special Issue Zebrafish as a Model in Human Disease 2.0)
►▼ Show Figures

Figure 1

Review

Jump to: Research

20 pages, 1780 KiB  
Review
Inflammation in Development and Aging: Insights from the Zebrafish Model
by Marta Mastrogiovanni, Francisco Juan Martínez-Navarro, Teresa V. Bowman and María L. Cayuela
Int. J. Mol. Sci. 2024, 25(4), 2145; https://doi.org/10.3390/ijms25042145 - 10 Feb 2024
Viewed by 1901
Abstract
Zebrafish are an emergent animal model to study human diseases due to their significant genetic similarity to humans, swift development, and genetic manipulability. Their utility extends to the exploration of the involvement of inflammation in host defense, immune responses, and tissue regeneration. Additionally, [...] Read more.
Zebrafish are an emergent animal model to study human diseases due to their significant genetic similarity to humans, swift development, and genetic manipulability. Their utility extends to the exploration of the involvement of inflammation in host defense, immune responses, and tissue regeneration. Additionally, the zebrafish model system facilitates prompt screening of chemical compounds that affect inflammation. This study explored the diverse roles of inflammatory pathways in zebrafish development and aging. Serving as a crucial model, zebrafish provides insights into the intricate interplay of inflammation in both developmental and aging contexts. The evidence presented suggests that the same inflammatory signaling pathways often play instructive or beneficial roles during embryogenesis and are associated with malignancies in adults. Full article
(This article belongs to the Special Issue Zebrafish as a Model in Human Disease 2.0)
►▼ Show Figures

Figure 1

15 pages, 1341 KiB  
Review
Zebrafish as an Emerging Model for Sarcopenia: Considerations, Current Insights, and Future Directions
by Santiago Callegari, Foad Mirzaei, Lila Agbaria, Sanobar Shariff, Burhan Kantawala, Desmond Moronge and Brian M. O. Ogendi
Int. J. Mol. Sci. 2023, 24(23), 17018; https://doi.org/10.3390/ijms242317018 - 30 Nov 2023
Viewed by 1100
Abstract
Sarcopenia poses a significant challenge to public health and can severely impact the quality of life of aging populations. Despite extensive efforts to study muscle degeneration using traditional animal models, there is still a lack of effective diagnostic tools, precise biomarkers, and treatments [...] Read more.
Sarcopenia poses a significant challenge to public health and can severely impact the quality of life of aging populations. Despite extensive efforts to study muscle degeneration using traditional animal models, there is still a lack of effective diagnostic tools, precise biomarkers, and treatments for sarcopenia. Zebrafish models have emerged as powerful tools in biomedical research, providing unique insights into age-related muscle disorders like sarcopenia. The advantages of using zebrafish models include their rapid growth outside of the embryo, optical transparency during early developmental stages, high reproductive potential, ease of husbandry, compact size, and genetic tractability. By deepening our understanding of the molecular processes underlying sarcopenia, we may develop novel diagnostic tools and effective treatments that can improve the lives of aging individuals affected by this condition. This review aims to explore the unique advantages of zebrafish as a model for sarcopenia research, highlight recent breakthroughs, outline potential avenues for future investigations, and emphasize the distinctive contributions that zebrafish models offer. Our research endeavors to contribute significantly to address the urgent need for practical solutions to reduce the impact of sarcopenia on aging populations, ultimately striving to enhance the quality of life for individuals affected by this condition. Full article
(This article belongs to the Special Issue Zebrafish as a Model in Human Disease 2.0)
►▼ Show Figures

Figure 1

29 pages, 1071 KiB  
Review
Assessing Drug Administration Techniques in Zebrafish Models of Neurological Disease
by Victoria Chaoul, Emanuel-Youssef Dib, Joe Bedran, Chakib Khoury, Omar Shmoury, Frédéric Harb and Jihane Soueid
Int. J. Mol. Sci. 2023, 24(19), 14898; https://doi.org/10.3390/ijms241914898 - 4 Oct 2023
Cited by 2 | Viewed by 3221
Abstract
Neurological diseases, including neurodegenerative and neurodevelopmental disorders, affect nearly one in six of the world’s population. The burden of the resulting deaths and disability is set to rise during the next few decades as a consequence of an aging population. To address this, [...] Read more.
Neurological diseases, including neurodegenerative and neurodevelopmental disorders, affect nearly one in six of the world’s population. The burden of the resulting deaths and disability is set to rise during the next few decades as a consequence of an aging population. To address this, zebrafish have become increasingly prominent as a model for studying human neurological diseases and exploring potential therapies. Zebrafish offer numerous benefits, such as genetic homology and brain similarities, complementing traditional mammalian models and serving as a valuable tool for genetic screening and drug discovery. In this comprehensive review, we highlight various drug delivery techniques and systems employed for therapeutic interventions of neurological diseases in zebrafish, and evaluate their suitability. We also discuss the challenges encountered during this process and present potential advancements in innovative techniques. Full article
(This article belongs to the Special Issue Zebrafish as a Model in Human Disease 2.0)
►▼ Show Figures

Figure 1

23 pages, 3668 KiB  
Review
Imaging Approaches to Investigate Pathophysiological Mechanisms of Brain Disease in Zebrafish
by Lapo Turrini, Lorenzo Roschi, Giuseppe de Vito, Francesco Saverio Pavone and Francesco Vanzi
Int. J. Mol. Sci. 2023, 24(12), 9833; https://doi.org/10.3390/ijms24129833 - 7 Jun 2023
Cited by 2 | Viewed by 1861
Abstract
Zebrafish has become an essential model organism in modern biomedical research. Owing to its distinctive features and high grade of genomic homology with humans, it is increasingly employed to model diverse neurological disorders, both through genetic and pharmacological intervention. The use of this [...] Read more.
Zebrafish has become an essential model organism in modern biomedical research. Owing to its distinctive features and high grade of genomic homology with humans, it is increasingly employed to model diverse neurological disorders, both through genetic and pharmacological intervention. The use of this vertebrate model has recently enhanced research efforts, both in the optical technology and in the bioengineering fields, aiming at developing novel tools for high spatiotemporal resolution imaging. Indeed, the ever-increasing use of imaging methods, often combined with fluorescent reporters or tags, enable a unique chance for translational neuroscience research at different levels, ranging from behavior (whole-organism) to functional aspects (whole-brain) and down to structural features (cellular and subcellular). In this work, we present a review of the imaging approaches employed to investigate pathophysiological mechanisms underlying functional, structural, and behavioral alterations of human neurological diseases modeled in zebrafish. Full article
(This article belongs to the Special Issue Zebrafish as a Model in Human Disease 2.0)
►▼ Show Figures

Figure 1

Show export options expand_more Show export options expand_less
Select all
Export citation of selected articles as:
 
Displaying articles 1-11
Back to TopTop