ArticleGenetics2013
The kinesin-3, unc-104 regulates dendrite morphogenesis and synaptic development in Drosophila.
Article in Genetics, 2013. The graph could read no effect estimate from its abstract, so it casts no vote on the map. Cited by 24 papers.
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Who cites it
24 citing papers in PubMed, 48 citations in OpenAlex.
- Motor Protein Disruption Critically Alters Organelle Trafficking and Excitation-Contraction Coupling.eNeuro · 2026Article
- Dopamine neuron specific RNA-sequencing reveals Neprilysin 1 acts downstream of the cohesin complex to suppress learning.Communications biology · 2026Article
- Soma-localized Rab39 inhibits synaptic autophagy by controlling trafficking of Atg9 vesicles.The EMBO journal · 2025Article
- Myosin 15 participates in assembly and remodeling of the presynapse.The Journal of cell biology · 2025Article
- Presynaptic Precursor Vesicles-Cargo, Biogenesis, and Kinesin-Based Transport across Species.Cells · 2023Review
- Molecular Logic of Synaptic Diversity BetweenbioRxiv : the preprint server for biology · 2023Article
- Autophagy inhibition rescues structural and functional defects caused by the loss of mitochondrial chaperoneAutophagy · 2021Article
- Recent Advances inInternational journal of molecular sciences · 2020Review
- Spatial control of membrane traffic in neuronal dendrites.Molecular and cellular neurosciences · 2020Review
- Pseudorabies Virus Infection Accelerates Degradation of the Kinesin-3 Motor KIF1A.Journal of virology · 2020Article
- Stromalin Constrains Memory Acquisition by Developmentally Limiting Synaptic Vesicle Pool Size.Neuron · 2019Article
- GDAP2 mutations implicate susceptibility to cellular stress in a new form of cerebellar ataxia.Brain : a journal of neurology · 2018Article
- Prd1 associates with the clathrin adaptor α-Adaptin and the kinesin-3 Imac/Unc-104 to govern dendrite pruning in Drosophila.PLoS biology · 2018Article
- A method for estimating relative changes in the synaptic density in Drosophila central nervous system.BMC neuroscience · 2018Article
- Article
- Intracellular Cargo Transport by Kinesin-3 Motors.Biochemistry. Biokhimiia · 2017Review
- The KIF1A homolog Unc-104 is important for spontaneous release, postsynaptic density maturation and perisynaptic scaffold organization.Scientific reports · 2017Article
- The cellular mechanisms that maintain neuronal polarity.Nature reviews. Neuroscience · 2016Review
- The Drosophila KIF1A Homolog unc-104 Is Important for Site-Specific Synapse Maturation.Frontiers in cellular neuroscience · 2016Article
- Nonfunctional ingestion of plant miRNAs in silkworm revealed by digital droplet PCR and transcriptome analysis.Scientific reports · 2015Article
Corrections and comments
PubMed lists nothing against this paper. Absence here is not a guarantee, only a check that was made.
Authors and funding
5 authors at 3 institutions in 2 countries.
Funding
Abstract
Kinesin-based transport is important for synaptogenesis, neuroplasticity, and maintaining synaptic function. In an anatomical screen of neurodevelopmental mutants, we identified the exchange of a conserved residue (R561H) in the forkhead-associated domain of the kinesin-3 family member Unc-104/KIF1A as the genetic cause for defects in synaptic terminal- and dendrite morphogenesis. Previous structure-based analysis suggested that the corresponding residue in KIF1A might be involved in stabilizing the activated state of kinesin-3 dimers. Herein we provide the first in vivo evidence for the functional importance of R561. The R561H allele (unc-104(bris)) is not embryonic lethal, which allowed us to investigate consequences of disturbed Unc-104 function on postembryonic synapse development and larval behavior. We demonstrate that Unc-104 regulates the reliable apposition of active zones and postsynaptic densities, possibly by controlling site-specific delivery of its cargo. Next, we identified a role for Unc-104 in restraining neuromuscular junction growth and coordinating dendrite branch morphogenesis, suggesting that Unc-104 is also involved in dendritic transport. Mutations in KIF1A/unc-104 have been associated with hereditary spastic paraplegia and hereditary sensory and autonomic neuropathy type 2. However, we did not observe synapse retraction or dystonic posterior paralysis. Overall, our study demonstrates the specificity of defects caused by selective impairments of distinct molecular motors and highlights the critical importance of Unc-104 for the maturation of neuronal structures during embryonic development, larval synaptic terminal outgrowth, and dendrite morphogenesis.
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Registered trials
Read under generation 80e0d062 · epoch 390. Bibliography from PubMed, PubMed Central and OpenAlex; grants from NIH RePORTER; trial links from ClinicalTrials.gov; estimates, votes and beliefs from the OpenQuestion graph.