Morphological, behavioral and cellular analyses revealed different phenotypes in Wolfram syndrome wfs1a and wfs1b zebrafish mutant lines DOI Creative Commons
Lucie Crouzier, Élodie M. Richard,

Camille Diez

et al.

Human Molecular Genetics, Journal Year: 2022, Volume and Issue: 31(16), P. 2711 - 2727

Published: March 17, 2022

Wolfram syndrome (WS) is a rare genetic disease characterized by diabetes, optic atrophy and deafness. Patients die at 35 years of age, mainly from respiratory failure or dysphagia. Unfortunately, there no treatment to block the progression symptoms an urgent need for adequate research models. Here, we report on phenotypical characterization two loss-of-function zebrafish mutant lines: wfs1aC825X wfs1bW493X. We observed that wfs1a deficiency altered size ear retina fish. also documented decrease in expression level unfolded protein response (UPR) genes basal condition stress condition, i.e. after tunicamycin treatment. Interestingly, both mutants lead their visual function measured behaviorally. These deficits were associated with UPR conditions. basal, ATP-linked maximal mitochondrial respirations transiently decreased wfs1b mutant. Taken together, these lines highlight critical role UPR, physiology. models will be useful tools better understand cellular Wfs1 develop novel therapeutic approaches WS.

Language: Английский

Unraveling circuits of visual perception and cognition through the superior colliculus DOI Creative Commons
Michele A. Basso, Martha E. Bickford, Jianhua Cang

et al.

Neuron, Journal Year: 2021, Volume and Issue: 109(6), P. 918 - 937

Published: Feb. 6, 2021

The superior colliculus is a conserved sensorimotor structure that integrates visual and other sensory information to drive reflexive behaviors. Although the evidence for this strong compelling, number of experiments reveal role in behaviors usually associated with cerebral cortex, such as attention decision-making. Indeed, addition collicular outputs targeting brainstem regions controlling movements, also has ascending projections linking it forebrain structures including basal ganglia amygdala, highlighting fact colliculus, its vast inputs outputs, can influence processing throughout neuraxis. Today, modern molecular genetic methods combined sophisticated behavioral assessments have potential make significant breakthroughs our understanding evolution conservation neuronal cell types circuits give rise simple complex

Language: Английский

Citations

171

The tectum/superior colliculus as the vertebrate solution for spatial sensory integration and action DOI Creative Commons
Tadashi Isa, Emmanuel Márquez-Legorreta, Sten Grillner

et al.

Current Biology, Journal Year: 2021, Volume and Issue: 31(11), P. R741 - R762

Published: June 1, 2021

The superior colliculus, or tectum in the case of non-mammalian vertebrates, is a part brain that registers events surrounding space, often through vision and hearing, but also electrosensation, infrared detection, other sensory modalities diverse vertebrate lineages. This information used to form maps space positions different salient stimuli relation individual. are arranged layers with visual input uppermost layer, senses deeper positions, spatially aligned motor map deepest layer. Here, we will review organization intrinsic function tectum/superior colliculus processed within tectal circuits. We discuss tectal/superior outputs conveyed directly downstream circuits via thalamus cortical areas control various aspects behavior. evolutionarily conserved among all tailored specialties each lineage, its roles have shifted emergence cerebral cortex mammals. illustrate both divergent properties evolution by comparing processing lampreys belonging oldest group extant larval zebrafish, rodents, vertebrates including primates.

Language: Английский

Citations

142

Transcriptomic neuron types vary topographically in function and morphology DOI Creative Commons
Inbal Shainer, Johannes M. Kappel,

Eva Laurell

et al.

Nature, Journal Year: 2025, Volume and Issue: unknown

Published: Feb. 12, 2025

Abstract Neuronal phenotypic traits such as morphology, connectivity and function are dictated, to a large extent, by specific combination of differentially expressed genes. Clusters neurons in transcriptomic space correspond distinct cell types some cases—for example, Caenorhabditis elegans 1 retinal ganglion cells 2–4 —have been shown share morphology function. The zebrafish optic tectum is composed spatial array that transforms visual inputs into motor outputs. Although the visuotopic map continuous, subregions functionally specialized 5,6 . Here, uncover cell-type architecture tectum, we transcriptionally profiled its neurons, revealing more than 60 organized anatomical layers. We measured responses thousands tectal two-photon calcium imaging matched them with their transcriptional profiles. Furthermore, characterized morphologies identified using transgenic lines. Notably, found similar can diverge shape, responses. Incorporating coordinates within volume revealed morphologically defined subclusters individual clusters. Our findings demonstrate extrinsic, position-dependent factors expand repertoire genetically neurons.

Language: Английский

Citations

2

Molecular classification of zebrafish retinal ganglion cells links genes to cell types to behavior DOI
Yvonne Kölsch, Joshua Hahn, Anna Sappington

et al.

Neuron, Journal Year: 2020, Volume and Issue: 109(4), P. 645 - 662.e9

Published: Dec. 23, 2020

Language: Английский

Citations

79

The Superior Colliculus: Cell Types, Connectivity, and Behavior DOI Creative Commons
Xue Liu,

Hongren Huang,

Terrance P. Snutch

et al.

Neuroscience Bulletin, Journal Year: 2022, Volume and Issue: 38(12), P. 1519 - 1540

Published: April 28, 2022

Abstract The superior colliculus (SC), one of the most well-characterized midbrain sensorimotor structures where visual, auditory, and somatosensory information are integrated to initiate motor commands, is highly conserved across vertebrate evolution. Moreover, cell-type-specific SC neurons integrate afferent signals within local networks generate defined output related innate cognitive behaviors. This review focuses on recent progress in understanding phenotypic diversity amongst their intrinsic circuits long-projection targets. We further describe relevant neural specific cell types relation behavioral outputs functions. systematic delineation organization, types, connections put into context species as these depend upon laminar architecture. we focus circuitry involving saccadic eye movement, Overall, provides insight functioning represents a basis for pathology associated with dysfunction.

Language: Английский

Citations

55

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

Precise visuomotor transformations underlying collective behavior in larval zebrafish DOI Creative Commons
Roy Harpaz,

Minh Nguyet Nguyen,

Armin Bahl

et al.

Nature Communications, Journal Year: 2021, Volume and Issue: 12(1)

Published: Nov. 12, 2021

Abstract Complex schooling behaviors result from local interactions among individuals. Yet, how sensory signals neighbors are analyzed in the visuomotor stream of animals is poorly understood. Here, we studied aggregation behavior larval zebrafish and found that over development larvae transition overdispersed groups to tight shoals. Using a virtual reality assay, characterized algorithms fish use transform visual inputs into movement decisions. We young turn away by integrating averaging retina-wide occupancy within each eye, using winner-take-all strategy for binocular integration. As mature, their responses expand include attraction neighbors, which based on similar model simulations, show observed accurately predict group structure development. These findings allow us make testable predictions regarding neuronal circuits underlying collective zebrafish.

Language: Английский

Citations

42

Neural mechanisms to exploit positional geometry for collision avoidance DOI Creative Commons
Ryosuke Tanaka, Damon A. Clark

Current Biology, Journal Year: 2022, Volume and Issue: 32(11), P. 2357 - 2374.e6

Published: May 3, 2022

Language: Английский

Citations

30

From perception to behavior: The neural circuits underlying prey hunting in larval zebrafish DOI Creative Commons
Shuyu Zhu, Geoffrey J. Goodhill

Frontiers in Neural Circuits, Journal Year: 2023, Volume and Issue: 17

Published: Feb. 1, 2023

A key challenge for neural systems is to extract relevant information from the environment and make appropriate behavioral responses. The larval zebrafish offers an exciting opportunity studying these sensing processes sensory-motor transformations. Prey hunting instinctual behavior of that requires brain combine different attributes sensory input form motor outputs. Due its small size transparency allows optical recording whole-brain activity reveal mechanisms involved in prey capture. In this review we discuss how visual identify locate prey, circuits governing generation commands response can be modulated by internal states experience, some outstanding questions field.

Language: Английский

Citations

18

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

8