Zebrafish Models of Autosomal Dominant Ataxias DOI Creative Commons
Ana Quelle‐Regaldie, Daniel Sobrido‐Cameán, Antón Barreiro‐Iglesias

et al.

Cells, Journal Year: 2021, Volume and Issue: 10(2), P. 421 - 421

Published: Feb. 17, 2021

Hereditary dominant ataxias are a heterogeneous group of neurodegenerative conditions causing cerebellar dysfunction and characterized by progressive motor incoordination. Despite many efforts put into the study these diseases, there no effective treatments yet. Zebrafish models widely used to characterize neuronal disorders due its conserved vertebrate genetics that easily support genetic edition their optic transparency allows observing intact CNS connections. In addition, small size external fertilization help develop high throughput assays candidate drugs. Here, we discuss contributions zebrafish defining phenotypes, function, behavior possible treatments. review created for X-linked repeat expansion diseases X-fragile/fragile-X tremor ataxia. Most reviewed here presented damage locomotor deficits. However, is generalized lack adult heterozygous knock-in available diseases. The helped elucidate gene function mechanisms cause damage. future, application new techniques would more accurate ataxias.

Language: Английский

Brain-wide visual habituation networks in wild type and fmr1 zebrafish DOI Creative Commons
Emmanuel Márquez-Legorreta, Lena Constantin,

Marielle Piber

et al.

Nature Communications, Journal Year: 2022, Volume and Issue: 13(1)

Published: Feb. 16, 2022

Abstract Habituation is a form of learning during which animals stop responding to repetitive stimuli, and deficits in habituation are characteristic several psychiatric disorders. Due technical challenges, the brain-wide networks mediating poorly understood. Here we report calcium imaging larval zebrafish repeated visual looming stimuli. We show that different functional categories loom-sensitive neurons located locations throughout brain, both properties their resulting behavior can be modulated by stimulus saliency timing. Using graph theory, identify circuit habituates minimally, moderately habituating midbrain population proposed mediate sensorimotor transformation, downstream elements responsible for higher order representations delivery behavior. Zebrafish larvae carrying mutation fmr1 gene have systematic shift toward sustained premotor activity this network, slower behavioral habituation.

Language: Английский

Citations

59

Whole-brain imaging of freely-moving zebrafish DOI Creative Commons

Hamid Hasani,

Jipeng Sun, Shuyu Zhu

et al.

Frontiers in Neuroscience, Journal Year: 2023, Volume and Issue: 17

Published: April 17, 2023

One of the holy grails neuroscience is to record activity every neuron in brain while an animal moves freely and performs complex behavioral tasks. While important steps forward have been taken recently large-scale neural recording rodent models, single resolution across entire mammalian remains elusive. In contrast larval zebrafish offers great promise this regard. Zebrafish are a vertebrate model with substantial homology brain, but their transparency allows whole-brain recordings genetically-encoded fluorescent indicators at single-neuron using optical microscopy techniques. Furthermore begin show repertoire natural behavior from early age, including hunting small, fast-moving prey visual cues. Until work address bases these behaviors mostly relied on assays where fish was immobilized under microscope objective, stimuli such as were presented virtually. However significant progress has made developing imaging techniques for which not immobilized. Here we discuss recent advances, focusing particularly based light-field microscopy. We also draw attention several outstanding issues remain be addressed increase ecological validity results obtained.

Language: Английский

Citations

35

The Visual Systems of Zebrafish DOI
Herwig Baier, Ethan K. Scott

Annual Review of Neuroscience, Journal Year: 2024, Volume and Issue: 47(1), P. 255 - 276

Published: April 25, 2024

The zebrafish visual system has become a paradigmatic preparation for behavioral and systems neuroscience. Around 40 types of retinal ganglion cells (RGCs) serve as matched filters stimulus features, including light, optic flow, prey, objects on collision course. RGCs distribute their signals via axon collaterals to 12 retinorecipient areas in forebrain midbrain. major visuomotor hub, the tectum, harbors nine RGC input layers that combine information multiple features. retinotopic map tectum is locally adapted scene statistics subfield–specific demands. Tectal projections premotor centers are topographically organized according commands. known connectivity more than 20 processing streams allows us dissect cellular basis elementary perceptual cognitive functions. Visually evoked responses, such prey capture or loom avoidance, controlled by dedicated multistation pathways that—at least larva—resemble labeled lines. This architecture serves neuronal code's purpose driving adaptive behavior.

Language: Английский

Citations

9

From macro to micro: De novo genomes of Aedes mosquitoes enable comparative genomics among close and distant relatives DOI Creative Commons
Gen Morinaga, Darío Balcazar, Athanase Badolo

et al.

bioRxiv (Cold Spring Harbor Laboratory), Journal Year: 2025, Volume and Issue: unknown

Published: Jan. 16, 2025

The yellow fever mosquito (Aedes aegypti) is an organism of high medical importance because it the primary vector for diseases such as fever, Zika, dengue, and chikungunya. Its has made a subject numerous efforts to understand their biology. One effort, was development high-quality reference genome (AaegL5). However, this sourced from highly inbred laboratory strain with unknown geographic origin. Thus, not representative wild mosquito, let alone one its native range in sub-Saharan Africa. To better genetic architecture Ae. aegypti sister species, we developed two de novo chromosome-scale genomes sequences single individuals: formosus (Aaf) Burkina Faso mascarensis (Am) Mauritius. Both exhibit contiguity gene completeness, comparable AaegL5. While Aaf exhibits degree synteny AaegL5, also several large inversions. We further conducted comparative genomic analyses using our other publicly available culicid find extensive chromosomal rearrangements between major lineages. Overrepresentation analysis expanded genes Aaf, Am revealed that while overarching category have are similar, specific differ. Our findings elucidate novel insights into chromosome evolution at both microevolutionary macroevolutionary scales. resources present additions arsenal biologists understanding biology evolution.

Language: Английский

Citations

1

Calcium Imaging and the Curse of Negativity DOI Creative Commons
Gilles Vanwalleghem, Lena Constantin, Ethan K. Scott

et al.

Frontiers in Neural Circuits, Journal Year: 2021, Volume and Issue: 14

Published: Jan. 6, 2021

The imaging of neuronal activity using calcium indicators has become a staple modern neuroscience. However, without ground truths, there is real risk missing significant portion the responses. Here, we show that common assumption, non-negativity responses as detected by indicators, biases all levels frequently used analytical methods for these data. From extraction meaningful fluorescence changes to spike inference and analysis inferred spikes, each step risks because assumption non-negativity. We first negative deviations from baseline can exist in activity. Then, use simulated data test three popular algorithms image analysis, CaImAn, suite2p, CellSort, finding suite2p may be best suited large datasets. also tested included Cellsort, well dedicated MLspike CASCADE, found have limitations dealing with inhibited neurons. Among algorithms, FOOPSI, performed on neurons, but even this algorithm spurious spikes upon return signal baseline. As such, new approaches will needed before sensitively accurately further suggest avoiding that, assuming non-negativity, ignore Instead, exploratory step, k-means or PCA example, detect whether are present. Taking steps ensure inhibition, excitation,

Language: Английский

Citations

44

Diving into the zebrafish brain: exploring neuroscience frontiers with genetic tools, imaging techniques, and behavioral insights DOI Creative Commons
Olga Doszyn, Tomasz Dulski, Justyna Zmorzyńska

et al.

Frontiers in Molecular Neuroscience, Journal Year: 2024, Volume and Issue: 17

Published: March 12, 2024

The zebrafish ( Danio rerio ) is increasingly used in neuroscience research. Zebrafish are relatively easy to maintain, and their high fecundity makes them suitable for high-throughput experiments. Their small, transparent embryos larvae allow microscopic imaging of the developing brain. also share a degree genetic similarity with humans, amenable manipulation techniques, such as gene knockdown, knockout, or knock-in, which allows researchers study role specific genes relevant human brain development, function, disease. can serve model behavioral studies, including locomotion, learning, social interactions. In this review, we present state-of-the-art methods function zebrafish, tools labeling single neurons neuronal circuits, live neural activity, synaptic dynamics protein interactions brain, optogenetic manipulation, use virtual reality technology testing. We highlight potential research, especially regarding genetic-based disorders discuss its certain limitations model.

Language: Английский

Citations

8

Brain-Wide Mapping of Water Flow Perception in Zebrafish DOI Creative Commons
Gilles Vanwalleghem,

Kevin Schuster,

Michael A. Taylor

et al.

Journal of Neuroscience, Journal Year: 2020, Volume and Issue: 40(21), P. 4130 - 4144

Published: April 10, 2020

Information about water flow, detected by lateral line organs, is critical to the behavior and survival of fish amphibians. While certain aspects flow processing have been revealed through electrophysiology, we lack a comprehensive description neurons that respond network they form. Here, use brain-wide calcium imaging in combination with microfluidic stimulation map out, at cellular resolution, neuronal responses involved perceiving information larval zebrafish. We find diverse array responding head-to-tail (h-t) tail-to-head (t-h) or both. Early this pathway, ganglia, almost exclusively simple presence h-t t-h but later includes specifically onset, representing accumulated displacement during stimulus, encoding speed flow. The reporting on these more nuanced details are located across numerous brain regions, including some not previously implicated processing. A graph theory-based analysis shows majority dedicated detection, reinforced our finding like velocity total only encoded for direction. results represent first important modality, provide departure point detailed studies network. SIGNIFICANCE STATEMENT In aquatic animals, detecting stimuli, networks interpret remain mysterious. imaged activity individual entire brains zebrafish, revealing all response types their locations as occurs. others attuned direction, speed, duration has passed stimulus. With information, modeled underlying network, describing system its simulating motion rudimentary

Language: Английский

Citations

47

Neural connections of the torus semicircularis in the adult Zebrafish DOI Creative Commons
Julián Yáñez,

Maider Hernández Eguiguren,

Ramón Anadón

et al.

The Journal of Comparative Neurology, Journal Year: 2024, Volume and Issue: 532(1)

Published: Jan. 1, 2024

Abstract The torus semicircularis (TS) of teleosts is a key midbrain center the lateral line and acoustic sensory systems. To characterize TS in adult zebrafish, we studied their connections using carbocyanine tracers applied to other related nuclei tracts. Two main nuclei, central ventrolateral, were differentiable by afferent connections. From TS, (TSc) numerous toropetal cells labeled bilaterally several primary octaval (anterior, magnocellular, descending, posterior nuclei), secondary nucleus, caudal octavolateralis perilemniscular region. In midbrain, contralateral TSc. diencephalon, from TSc observed ipsilaterally medial prethalamic nucleus periventricular tubercle nucleus. neurons also hypothalamic anterior tuberal (ATN) parvicellular preoptic but not telencephalon. Tracer application revealed projections ventrolateral (TSvl), whereas tracer fibers rostral sends ascending ipsilateral preglomerular region that, turn, projects pallium. Application DiI optic tectum TSvl, ATN These results reveal that TSvl are mainly with mechanosensory centers, respectively, they show different higher order

Language: Английский

Citations

5

Sleep Problems in Preschoolers With Autism Spectrum Disorder Are Associated With Sensory Sensitivities and Thalamocortical Overconnectivity DOI Creative Commons
Annika C. Linke, Bosi Chen, Lindsay Olson

et al.

Biological Psychiatry Cognitive Neuroscience and Neuroimaging, Journal Year: 2021, Volume and Issue: 8(1), P. 21 - 31

Published: July 31, 2021

Projections between the thalamus and sensory cortices are established early in development play an important role regulating sleep as well relaying information to cortex. Atypical thalamocortical functional connectivity frequently observed children with autism spectrum disorder (ASD) might therefore be linked problems common ASD. Here, we investigated relationship auditory-thalamic measured during natural magnetic resonance imaging, problems, sound sensitivities 70 toddlers preschoolers (1.5–5 years old) ASD compared a matched group of 46 typically developing children. In ASD, were positively correlated, increased latency was associated overconnectivity auditory cortex subsample high-quality imaging data (n = 29). addition, blood oxygen level–dependent signal amplitude elevated potentially reflecting reduced gating or lack habituation sleep. These findings indicate that atypical can detected may crucial

Language: Английский

Citations

32

A Systematic Review of Brainstem Contributions to Autism Spectrum Disorder DOI Creative Commons

Ala Seif,

Carly Shea,

Susanne Schmid

et al.

Frontiers in Integrative Neuroscience, Journal Year: 2021, Volume and Issue: 15

Published: Nov. 1, 2021

Autism spectrum disorder (ASD) is a neurodevelopmental that affects one in 66 children Canada. The contributions of changes the cortex and cerebellum to autism have been studied for decades. However, our understanding brainstem has only started emerge more recently. Disruptions sensory processing, startle response, filtering, sensorimotor gating, multisensory integration sleep are all features ASD processes which involved. In addition, preliminary research into contribution emphasizes importance developmental timeline rather than just mature brainstem. Therefore, purpose this systematic review compile histological, behavioral, neuroimaging, electrophysiological evidence from human animal studies about their functional implications autism. Moreover, due nature autism, pays attention atypical development compares findings based on age. Overall, there an important role disruptions ASD, but still need examine across life span, infancy adulthood could lead way early diagnosis possibly treatment ASD.

Language: Английский

Citations

32