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

и другие.

Опубликована: Май 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.

Язык: 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

и другие.

Antioxidants, Год журнала: 2021, Номер 10(12), С. 1948 - 1948

Опубликована: Дек. 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.

Язык: Английский

Процитировано

62
Spatiotemporally resolved transcriptomics reveals the cellular dynamics of human retinal development DOI Creative Commons
Jinglong Zhang, Jiao Wang,

Qiongjie Zhou

и другие.

Nature Communications, Год журнала: 2025, Номер 16(1)

Опубликована: Март 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.

Язык: Английский

Процитировано

1
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

и другие.

Journal of Medical Genetics, Год журнала: 2024, Номер unknown, С. jmg - 109531

Опубликована: Янв. 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.

Язык: Английский

Процитировано

8
Theoretical prediction of broadband ambient light optogenetic vision restoration with ChRmine and its mutants DOI Creative Commons
Himanshu Bansal,

Gur Pyari,

Sukhdev Roy

и другие.

Scientific Reports, Год журнала: 2024, Номер 14(1)

Опубликована: Май 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.

Язык: Английский

Процитировано

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

Silke Feil

и другие.

Cells, Год журнала: 2024, Номер 13(13), С. 1142 - 1142

Опубликована: Июль 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.

Язык: Английский

Процитировано

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

и другие.

Developmental Cell, Год журнала: 2024, Номер 59(21), С. 2897 - 2911.e6

Опубликована: Авг. 13, 2024

Язык: Английский

Процитировано

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

и другие.

Genes & Development, Год журнала: 2021, Номер 35(9-10), С. 677 - 691

Опубликована: Апрель 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.

Язык: Английский

Процитировано

22
NLRP3 and autophagy in retinal ganglion cell inflammation in age-related macular degeneration: potential therapeutic implications DOI Creative Commons

Xiaoli Wang,

Yun-Xia Gao,

Qiong-Zhen Yuan

и другие.

International Journal of Ophthalmology, Год журнала: 2024, Номер 17(8), С. 1531 - 1544

Опубликована: Июль 24, 2024

Retinal degenerative diseases were a large group of characterized by the primary death retinal ganglion cells (RGCs). Recent studies had shown an interaction between autophagy and nucleotide-binding oligomerization domain-like receptor 3 (NLRP3) inflammasomes, which may affect RGCs in diseases. The NLRP3 inflammasome was protein complex that, upon activation, produces caspase-1, mediating apoptosis promoting occurrence development Upregulated could inhibit while inhibited can promote leaded to accelerated emergence drusen lipofuscin deposition under neurosensory retina. activated further autophagy, thus forming vicious cycle that damage RGCs. This review discussed relationship its effects on age-related macular degeneration, providing new perspective direction for treatment

Язык: Английский

Процитировано

3
Ciliary biology intersects autism and congenital heart disease DOI Creative Commons
Nia Teerikorpi, Micaela Lasser, Sheng Wang

и другие.

bioRxiv (Cold Spring Harbor Laboratory), Год журнала: 2024, Номер unknown

Опубликована: Июль 31, 2024

ABSTRACT Autism spectrum disorder (ASD) commonly co-occurs with congenital heart disease (CHD), but the molecular mechanisms underlying this comorbidity remain unknown. Given that children CHD come to clinical attention by newborn period, understanding which variants carry ASD risk could provide an opportunity identify and treat individuals at high for developing far before typical age of diagnosis. Therefore, it is critical delineate subset genes most likely increase ASD. However, date there relatively limited overlap between confidence genes, suggesting alternative strategies prioritizing are necessary. Recent studies have shown gene perturbations dysregulate neural progenitor cell (NPC) biology. Thus, we hypothesized disrupt neurogenesis more Hence, performed in vitro pooled CRISPR interference (CRISPRi) screen NPC biology similarly genes. Overall, identified 45 strongly impact proliferation and/or survival NPCs. Moreover, observed a cluster physically interacting enriched ciliary Studying seven these evidence shared ( CEP290, CHD4, KMT2E, NSD1, OFD1, RFX3, TAOK1 ), observe perturbation significantly impacts primary cilia formation . While vivo investigation reveals previously unappreciated role motile development, supporting its prediction as gene. Together, our findings highlight set may underscore

Язык: Английский

Процитировано

3
Revealing the neurodevelopmental toxicity of face mask-derived microplastics to humans based on neural organoids DOI
Minghui Li, Xue Gao,

Yingying Lan

и другие.

Journal of Hazardous Materials, Год журнала: 2025, Номер unknown, С. 138084 - 138084

Опубликована: Март 1, 2025

Язык: Английский

Процитировано

0