FB2025_01 , released February 20, 2025
Allele: Dmel\Usp519-2
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General Information
Symbol
Dmel\Usp519-2
Species
D. melanogaster
Name
FlyBase ID
FBal0298995
Feature type
allele
Associated gene
Associated Insertion(s)
Carried in Construct
Also Known As
19-2
Key Links
Nature of the Allele
Caused by aberration
Cytology
Description

Imprecise excision of P{EPgy2}BtbVIIEY07802, resulting in deletion of a 1kb region including the shared promoter region of BtbVII and Usp5 and the predicted ATG codon of Usp5.

Mutations Mapped to the Genome
Curation Data
Type
Location
Additional Notes
References
Variant Molecular Consequences
Associated Sequence Data
DNA sequence
Protein sequence
 
Expression Data
Reporter Expression
Additional Information
Statement
Reference
 
Marker for
Reflects expression of
Reporter construct used in assay
Human Disease Associations
Disease Ontology (DO) Annotations
Models Based on Experimental Evidence ( 0 )
Disease
Evidence
References
Modifiers Based on Experimental Evidence ( 0 )
Disease
Interaction
References
Comments on Models/Modifiers Based on Experimental Evidence ( 0 )
 
Disease-implicated variant(s)
 
Phenotypic Data
Phenotypic Class
Phenotype Manifest In
Detailed Description
Statement
Reference

Usp5leon-1/Usp519-2 as well as Usp5leon-2/Usp519-2 mutant third instar larvae display neuromuscular junction (NMJ) defects and the synaptic boutons are aggregated and less numerous relative to wild-type controls, and in Usp5leon-2/Usp519-2 the total branch length is also significantly decreased. Muscle area is not different from controls.

Although the overall neuromuscular junction bouton area in either Usp519-2 homozygous or Usp5leon-1/Usp519-2 transheterozygous mutant larvae is comparable to controls, the area of the subsynaptic reticulum surrounding it is much larger and the average length of synaptic membranes is also increased, whereas the number of T-bars per bouton or the vesicle density is not significantly different from controls. The electrophysiological properties of the Usp519-2 and Usp5leon-1/Usp519-2 mutant NMJs are also perturbed: while both the frequency and amplitude of miniature excitatory junction current (mEJC) is not significantly different compared to Usp519-2 heterozygous controls, the EJC amplitude and quantal content is significantly reduced.

Usp5leon-1/Usp519-2 larvae appear normal in body size, but brain lobes and wing disks are smaller than normal. Larval growth is also slower compared to controls.

External Data
Interactions
Show genetic interaction network for Enhancers & Suppressors
Phenotypic Class
Phenotype Manifest In
Additional Comments
Genetic Interactions
Statement
Reference
Xenogenetic Interactions
Statement
Reference
Complementation and Rescue Data
Comments

The neuromuscular junction morphological defects (decreased total branch length, reduced number of boutons) observed in Usp5leon-1/Usp519-2 mutant third instar larvae are fully rescued by combination with Usp5T:Avic\GFP or by expression of Usp5Scer\UAS.T:Zzzz\FLAG driven by Scer\GAL4how-24B but not if driven by Scer\GAL4Toll-6-D42, expression of Usp5ED.Scer\UAS.T:Zzzz\FLAG fails to rescue the morphology regardless of the driver.

Usp5T:Avic\GFP and Usp5Scer\UAS.T:Zzzz\FLAG expression (with the Scer\GAL4how-24B driver and not with Scer\GAL4Toll-6-D42) also rescues the impaired electrophysiological properties at Usp519-2 or Usp5leon-1/Usp519-2 mutant neuromuscular junctions (reduced excitatory junction current amplitude and quantal content).

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Mutant
Wild-type
Stocks (0)
Notes on Origin
Discoverer
External Crossreferences and Linkouts ( 0 )
Synonyms and Secondary IDs (2)
Reported As
Symbol Synonym
Name Synonyms
Secondary FlyBase IDs
    References (2)