Axon arrival times and physical occupancy establish visual projection neuron integration on developing dendrites in the Drosophila optic glomeruli DOI Open Access
Brennan W. McFarland, HyoJong Jang, Natalie Smolin

et al.

Published: May 9, 2024

Behaviorally relevant, higher order representations of an animal’s environment are built from the convergence visual features encoded in early stages processing. Although developmental mechanisms that generate feature encoding channels circuits have been uncovered, relatively little is known about direct to enable appropriate integration into downstream circuits. Here we explore development a collision detection sensorimotor circuit Drosophila melanogaster , projection neurons (VPNs) onto dendrites large descending neuron, giant fiber (GF). We find VPNs different establish their respective territories on GF through sequential axon arrival during development. Physical occupancy, but not activity, important maintain territories. Ablation one VPN results expansion remaining and functional compensation enables retain responses ethologically relevant stimuli. observed using pupal electrophysiology preparation, appears after established, likely contributes later synapse assembly refinement. Our data highlight temporal for promote optic glomeruli, where connectivity resides, as powerful model investigating complex wiring programs plasticity.

Language: Английский

Role of Oxidative Stress in Ocular Diseases Associated with Retinal Ganglion Cells Degeneration DOI Creative Commons
Eugene Yu‐Chuan Kang, Pei‐Kang Liu, Yao‐Tseng Wen

et al.

Antioxidants, Journal Year: 2021, Volume and Issue: 10(12), P. 1948 - 1948

Published: Dec. 5, 2021

Ocular diseases associated with retinal ganglion cell (RGC) degeneration is the most common neurodegenerative disorder that causes irreversible blindness worldwide. It characterized by visual field defects and progressive optic nerve atrophy. The underlying pathophysiology mechanisms of RGC in several ocular remain largely unknown. RGCs are a population central nervous system neurons, their soma located retina long axons extend through to form distal terminals connections brain. Because this unique cytoarchitecture highly compartmentalized energy demand, mitochondrial-dependent for adenosine triphosphate (ATP) production. Recently, oxidative stress mitochondrial dysfunction have been found be principal as well other disorders. Here, we review role degenerations, including glaucoma, hereditary atrophy, inflammatory neuritis, ischemic neuropathy, traumatic drug toxicity. We also experimental approaches using animal models research on degeneration. Lastly, discuss application antioxidants potential future therapy degenerations.

Language: Английский

Citations

62
BTB domain mutations perturbing KCTD15 oligomerisation cause a distinctive frontonasal dysplasia syndrome DOI Creative Commons
Kerry A. Miller, David A. Cruz Walma, Daniel M. Pinkas

et al.

Journal of Medical Genetics, Journal Year: 2024, Volume and Issue: unknown, P. jmg - 109531

Published: Jan. 31, 2024

encodes an oligomeric BTB domain protein reported to inhibit neural crest formation through repression of Wnt/beta-catenin signalling, as well transactivation by TFAP2. Heterozygous missense variants in the closely related paralogue KCTD1 cause scalp-ear-nipple syndrome.

Language: Английский

Citations

8
Spatiotemporally resolved transcriptomics reveals the cellular dynamics of human retinal development DOI Creative Commons
Jinglong Zhang, Jiao Wang,

Qiongjie Zhou

et al.

Nature Communications, Journal Year: 2025, Volume and Issue: 16(1)

Published: March 7, 2025

The morphogenesis and cellular interactions in developing retina are incompletely characterized. full understanding needs a precise mapping of the gene expression with single-cell spatial resolution. Here, we present transcriptomic (ST) resource for human at six developmental stages. Combining data enables characterization cell-type-specific profiles distinct anatomical regions each stage, highlighting spatiotemporal dynamics composition during retinal development. All ST spots catalogued into consensus domains, which further associated to their specific signatures biological functions neuron eye We prioritize set critical regulatory genes transitions domains Differentially expressed from different diseases, indicating relevance clinical significance spatially defined expression. Finally, reconstruct communication networks, highlight ligand-receptor Overall, our study reports development, provides rich future studies on retinogenesis.

Language: Английский

Citations

1
Theoretical prediction of broadband ambient light optogenetic vision restoration with ChRmine and its mutants DOI Creative Commons
Himanshu Bansal,

Gur Pyari,

Sukhdev Roy

et al.

Scientific Reports, Journal Year: 2024, Volume and Issue: 14(1)

Published: May 21, 2024

Vision restoration is one of the most promising applications optogenetics. However, it limited due to poor-sensitivity, slow-kinetics and narrow band absorption spectra opsins. Here, a detailed theoretical study retinal ganglion neurons (RGNs) expressed with ChRmine, ReaChR, CoChR, CatCh their mutants, near monochromatic LEDs, broadband sunlight, halogen lamp, RGB LED light, pure white light sources has been presented. All opsins exhibit improved sensitivity larger photocurrent on illuminating compared LEDs. ChRmine allows firing at ambient sunlight (1.5 nW/mm2) (1.2 nW/mm2), which lowest among considered. The activation spectrum its mutants also useful restore color sensitivity. Although exhibits slower turn-off kinetics high-fidelity spikes can be evoked upto 50 Hz. This limit extends 80 Hz hsChRmine mutant although requires double irradiance ChRmine. present shows that allow RGNs for goggle-free optogenetic prostheses quality restored vision.

Language: Английский

Citations

6
Identification and Characterization of ATOH7-Regulated Target Genes and Pathways in Human Neuroretinal Development DOI Creative Commons
David Atac, Kevin Maggi,

Silke Feil

et al.

Cells, Journal Year: 2024, Volume and Issue: 13(13), P. 1142 - 1142

Published: July 3, 2024

The proneural transcription factor atonal basic helix–loop–helix 7 (ATOH7) is expressed in early progenitors the developing neuroretina. In vertebrates, this crucial for development of retinal ganglion cells (RGCs), as mutant animals show an almost complete absence RGCs, underdeveloped optic nerves, and aberrations vessel development. Human mutations are rare result autosomal recessive nerve hypoplasia (ONH) or severe vascular changes, diagnosed persistent hyperplasia primary vitreous (PHPVAR). To better understand role ATOH7 neuroretinal development, we created knockout eGFP-expressing reporter human induced pluripotent stem (hiPSCs), which were differentiated into early-stage organoids. Target loci regulated by identified Cleavage Under Targets Release Using Nuclease with sequencing (CUT&RUN-seq) differential expression RNA (RNA-seq) wildtype organoid-derived cells. Additionally, single-cell (scRNA-seq) was performed on whole organoids to identify cell type-specific genes. Mutant displayed substantial deficiency axon sprouting, reduction increase other types. We 469 differentially target genes, overrepresentation genes belonging development/guidance Notch signaling. Taken together, consolidate function guiding progenitor competence inducing RGC-specific while inhibiting fates. Furthermore, highlight candidate responsible ATOH7-associated retinovascular anomalies, sheds light potential future therapy targets related disorders.

Language: Английский

Citations

4
Developmental control of rod number via a light-dependent retrograde pathway from intrinsically photosensitive retinal ganglion cells DOI
Shane P. D’Souza, Brian A. Upton, Kiara C. Eldred

et al.

Developmental Cell, Journal Year: 2024, Volume and Issue: 59(21), P. 2897 - 2911.e6

Published: Aug. 13, 2024

Language: Английский

Citations

4
Building a circuit through correlated spontaneous neuronal activity in the developing vertebrate and invertebrate visual systems DOI Open Access
Ben Jiwon Choi, Yu‐Chieh Chen, Claude Desplan

et al.

Genes & Development, Journal Year: 2021, Volume and Issue: 35(9-10), P. 677 - 691

Published: April 22, 2021

During the development of vertebrate nervous systems, genetic programs assemble an immature circuit that is subsequently refined by neuronal activity evoked external stimuli. However, prior to sensory experience, intrinsic property developing system also triggers correlated network-level activity, with retinal waves in retina being best documented example. Spontaneous has been found visual Drosophila . Here, we compare spontaneous between mammalian and suggest emerging model for mechanistic functional studies activity.

Language: Английский

Citations

22
Revealing the neurodevelopmental toxicity of face mask-derived microplastics to humans based on neural organoids DOI
Minghui Li, Xue Gao,

Yingying Lan

et al.

Journal of Hazardous Materials, Journal Year: 2025, Volume and Issue: unknown, P. 138084 - 138084

Published: March 1, 2025

Language: Английский

Citations

0
Visual performance of single-vision contact lenses utilizing opaque, nonrefractive features for potential myopia management DOI
Daniel Tilia,

Jennie Diec,

Jennifer Sha

et al.

Optometry and Vision Science, Journal Year: 2025, Volume and Issue: unknown

Published: April 18, 2025

SIGNIFICANCE: Contact lenses (CLs) utilizing opaque, nonrefractive features may purposefully modulate retinal ganglion cell activity away from the baseline activity. This is a mechanism that reduce myopia progression. However, visual performance of CLs with opaque unknown. PURPOSE: study aimed to compare and binocular/accommodative function (test) against MiSight (control-1) single-vision (control-2) CLs. METHODS: was prospective, randomized, unmasked, cross-over where 35 myopic CL wearers (18 39 years) wore each design for at least 5 days. Visual subjectively assessed using 1 10 numeric ratings comprising clarity vision, lack ghosting, vision when driving, overall satisfaction, willingness purchase (yes/no: based on efficacy). acuity measurements comprised monocular binocular high low contrast 6 m, 70 40 cm. Binocular heterophorias 3 m Accommodative accommodative facility (MAF) cm dynamic response (AR: cm, cm). RESULTS: Test rated higher than control-1 (p<0.001) control-2 test (p≤0.0052) all subjective ratings. More participants were willing compared efficacy (p<0.001), while there no difference between either question (p>0.7). Both controls better acuity-based (p≤0.0013). MAF (p=0.010) not different (p>0.99). AR both (p<0.0001), (p=0.12). There differences or (p>0.1). CONCLUSIONS: Compared control-1, offered performance, proportion purchase, MAF. control-2, worse but different, comparable.

Language: Английский

Citations

0
Early visual experience elicits cellular and functional plasticity in the retina and alters behaviour DOI Creative Commons

Peter C. Reynolds,

Davide Marchi, Yan To Ling

et al.

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

Published: April 29, 2025

Abstract Our interaction with the surrounding environment shapes how our brain processes sensory information and drives adaptive behaviour. This plasticity allows to rewire in response specific experiences. For instance, early manipulation of visual inputs profoundly impacts plasticity, which is crucial for functions like size perception, object recognition, visuospatial processing. While neuronal has been detected target structures such as colliculus, thalamus, cortex, it remains unclear if retina, primary organ, undergoes significant plasticity. Here, we show that zebrafish retina demonstrates pronounced plastic transformations alterations during development, ultimately modifies detection oriented stimuli. We demonstrate orientation-selective amacrine cells undergo profound morphological changes animals exposed distinct environments development. further find functional output from altered a manner consistent are raised these persistent. Finally, tested virtual reality system exposure different their innate preference specifically patterns. findings unveil unique developmental form organ continuing structural consequences.

Language: Английский

Citations

0