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General Information
Symbol
Hsap\FUSP525L.10x.UAS.Tag:FLAG
Species
H. sapiens
Name
FlyBase ID
FBal0265764
Feature type
allele
Associated gene
Associated Insertion(s)
Carried in Construct
Allele class
Nature of the Allele
Allele class
Mutations Mapped to the Genome
 
Type
Location
Additional Notes
References
Associated Sequence Data
DNA sequence
Protein sequence
 
 
Progenitor genotype
Carried in construct
Cytology
Nature of the lesion
Statement
Reference

Ten copies of UAS regulatory sequences drive expression of a P525L mutant Hsap\FUS cDNA fused to an N-terminal Tag:FLAG epitope tag.

Allele components
Product class / Tool use(s)
Encoded product / tool
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 ( 1 )
Disease
Evidence
References
Modifiers Based on Experimental Evidence ( 1 )
Disease
Interaction
References
Comments on Models/Modifiers Based on Experimental Evidence ( 0 )
 
Phenotypic Data
Phenotypic Class
Phenotype Manifest In
Detailed Description
Statement
Reference

The expression of Hsap\FUSP525L.10x.Scer\UAS.T:Zzzz\FLAG under the control of Scer\GAL4Toll-6-D42 does not lead to any obvious defects in viability or third instar larval locomotion, but induces a significant and progressive decrease in adult locomotion, as compared to controls.

The expression of Hsap\FUSP525L.10x.Scer\UAS.T:Zzzz\FLAG under the control of Scer\GAL4CCAP.PI leads to a decrease in axonal transport of NPY-positive vesicles (i.e. a significant decrease in anterograde transport and a significant increase in the stationary fraction), but not of mitochondria, in third instar larval motor neurons, as compared to controls.

Expression of Hsap\FUSP525L.10x.Scer\UAS.T:Zzzz\FLAG in motor neurons under the control of Scer\GAL4D42 results in impaired larval locomotion, which is rescued by pioglitazone-treatment. However, pioglitazone-treatment is not effective in improving lifespan in adult transgenic animals.

Scer\GAL4VGlut-OK371>Hsap\FUSP525L.10x.Scer\UAS.T:Zzzz\FLAG-expressing animals display a reduction in the number of presynaptic active zones compared with wild-type.

Compared with wild-type, third instar larvae expressing Hsap\FUSP525L.10x.Scer\UAS.T:Zzzz\FLAG under the control of Scer\GAL4VGlut-OK371 display an approximately 25% reduction in evoked excitatory junction potential (EJP) amplitude. The reduction of quantal content in these animals reaches statistical significance.

Analysis of synaptic fatigue reveals that animals expressing Scer\GAL4VGlut-OK371>Hsap\FUSP525L.10x.Scer\UAS.T:Zzzz\FLAG fatigue faster than control animals.

Overexpression of Hsap\FUSP525L.10x.Scer\UAS.T:Zzzz\FLAG in motor neurons via Scer\GAL4RapGAP1-OK6 does not result in neuromuscular junction (NMJ) expansion.

Overexpression of Hsap\FUSP525L.10x.Scer\UAS.T:Zzzz\FLAG in neurons has no significant effect on locomotion.

Overexpression of Hsap\FUSP525L.10x.Scer\UAS.T:Zzzz\FLAG has no effect on longevity.

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

The expression of Hsap\FUSP525L.10x.Scer\UAS.T:Zzzz\FLAG under the control of Scer\GAL4Toll-6-D42 nearly fully suppresses the semi-lethality, the decreased third instar larval crawling capacity and the decreased adult locomotor capacity observed in caz383 homozygotes.

The expression of Hsap\FUSP525L.10x.Scer\UAS.T:Zzzz\FLAG under the control of Scer\GAL4CCAP.PI fully suppresses the decreased axonal transport of mitochondria, but not of (NPY-positive) vesicles, observed in caz383 homozygous third instar larval motor neurons.

Expression of Hsap\FUSP525L.10x.Scer\UAS.T:Zzzz\FLAG in the nervous system under the control of Scer\GAL4elav-C155 almost completely rescues the adult viability of males hemizygous for the 'caz1' mutant chromosome (Df(1)383 onto which a transgene containing CG32576+t5.0 has been recombined to provide CG32576[+] function) - the level of rescue seen with Hsap\FUSP525L.10x.Scer\UAS.T:Zzzz\FLAG is not significantly different from that seen when a transgene containing wild-type caz is expressed using the same driver.

Expression of Hsap\FUSP525L.10x.Scer\UAS.T:Zzzz\FLAG under the control of Scer\GAL4elav-C155 partially rescues the locomotor defects seen in caz1/Y adult males.

Expression of Hsap\FUSP525L.10x.Scer\UAS.T:Zzzz\FLAG under the control of Scer\GAL4elav-C155 does not rescue the the median lifespan of adult caz1/Y escapers, although maximum lifespan is increased.

Complementation and Rescue Data
Comments
Images (0)
Mutant
Wild-type
Stocks (0)
Notes on Origin
Discoverer
External Crossreferences and Linkouts ( 0 )
Synonyms and Secondary IDs (3)
Reported As
Symbol Synonym
Hsap\FUSP525L.10x.Scer\UAS.T:Zzzz\FLAG
Hsap\FUSP525L.10x.UAS.Tag:FLAG
Name Synonyms
Secondary FlyBase IDs
    References (6)