FlyBase Allele Report: Dmel\Usp5[leon-1]

General Information

Symbol

Dmel\Usp5^leon-1

Species

D. melanogaster

Name

FlyBase ID

FBal0298993

Feature type

allele

Associated gene

Dmel\Usp5

Associated Insertion(s)

Carried in Construct

Also Known As

leon¹

Key Links

Genomic Maps

JBrowse

Allele class

loss of function allele

Mutagen

ethyl methanesulfonate

Nature of the Allele

Allele class

loss of function allele

(Wang et al., 2014)

Mutagen

ethyl methanesulfonate

(Wang et al., 2014)

Progenitor genotype

Cytology

Description

Amino acid replacement: C224Y.

(Wang et al., 2014)

Mutations Mapped to the Genome

Curation Data

Type

Location

Additional Notes

References

point_mutation

3L:3,147,314..3,147,314 [-]

Nucleotide change:

G3147314A

Amino acid change:

C224Y | Usp5-PA; C224Y | Usp5-PB; C224Y | Usp5-PC

Reported amino acid change:

C224Y

Comment:

Site of nucleotide substitution in mutant inferred by FlyBase based on reported amino acid change.

(Wang et al., 2014)

Variant Molecular Consequences

Associated Sequence Data

DDBJ / EMBL / GenBank

DNA sequence

Protein sequence

UniProtKB/Swiss-Prot

UniProtKB/TrEMBL

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

intermediate neuronal progenitor (with Usp5^leon-2)

(Komori et al., 2025)

larval brain (with Usp5^19-2)

(Wang et al., 2014)

neuromuscular junction | third instar larval stage (with Usp5^19-2)

(Wang et al., 2017)

NMJ bouton | third instar larval stage (with Usp5^19-2)

(Wang et al., 2017)

subsynaptic reticulum | third instar larval stage (with Usp5^19-2)

(Wang et al., 2017)

synaptic membrane | third instar larval stage (with Usp5^19-2)

(Wang et al., 2017)

wing disc (with Usp5^19-2)

(Wang et al., 2014)

Detailed Description

Statement

Reference

Usp5^leon-1/Usp5^19-2 mutant third instar larvae display neuromuscular junction (NMJ) defects: the axonal terminals fail to extend (the total branch length is significantly decreased) and the synaptic boutons are aggregated and less numerous relative to wild-type controls (muscle area is not different from controls). Although the overall neuromuscular junction bouton area in Usp5^leon-1/Usp5^19-2 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 Usp5^leon-1/Usp5^19-2 NMJs are also perturbed: while both the frequency and amplitude of miniature excitatory junction current (mEJC) is not significantly different compared to Usp5^19-2 heterozygous controls, the EJC amplitude and quantal content is significantly reduced.

(Wang et al., 2017)

Usp5^leon-1/Usp5^19-2 larvae appear normal in body size, but brain lobes and wing disks are smaller than normal. Larval growth is also slower compared to controls.

(Wang et al., 2014)

External Data

Linkouts

Interactions

Show genetic interaction network for Enhancers & Suppressors

Phenotypic Class

Suppressed by

Statement

Reference

Usp5^leon-1, brat^DG19310/brat¹¹ has increased cell number | larval stage phenotype, suppressible by Scer\GAL4^GMR9D11/Usp5^UAS.Tag:FLAG

(Komori et al., 2025)

BacA\p35^UAS.cHa, Scer\GAL4^wor.PA, Scer\GAL80^ase.PN, Usp5^leon-1 has increased cell number | larval stage phenotype, suppressible by Usp5^UAS.Tag:FLAG, Scer\GAL4^wor.PA, Scer\GAL80^ase.PN

(Komori et al., 2025)

NOT suppressed by

Statement

Reference

BacA\p35^UAS.cHa, Scer\GAL4^wor.PA, Scer\GAL80^ase.PN, Usp5^leon-1 has increased cell number | larval stage phenotype, non-suppressible by Usp5^{ED.UAS.Tag:FLAG}, Scer\GAL4^wor.PA, Scer\GAL80^ase.PN

(Komori et al., 2025)

Enhancer of

Statement

Reference

Usp5^leon-2/Usp5^leon-1 is an enhancer of increased cell number | larval stage phenotype of brat^DG19310/brat¹¹

(Komori et al., 2025)

Usp5^leon-1/Usp5^leon-1 is an enhancer of increased cell number | larval stage phenotype of brat^DG19310/brat¹¹

(Komori et al., 2025)

Other

Statement

Reference

BacA\p35^UAS.cHa, Scer\GAL4^wor.PA, Scer\GAL80^ase.PN, Usp5^leon-1 has increased cell number | larval stage phenotype

(Komori et al., 2025)

Phenotype Manifest In

Suppressed by

Statement

Reference

Usp5^leon-1, brat^DG19310/brat¹¹ has type II neuroblast | increased number phenotype, suppressible by Scer\GAL4^GMR9D11/Usp5^UAS.Tag:FLAG

(Komori et al., 2025)

BacA\p35^UAS.cHa, Scer\GAL4^wor.PA, Scer\GAL80^ase.PN, Usp5^leon-1 has type II neuroblast | increased number phenotype, suppressible by Usp5^UAS.Tag:FLAG, Scer\GAL4^wor.PA, Scer\GAL80^ase.PN

(Komori et al., 2025)

NOT suppressed by

Statement

Reference

BacA\p35^UAS.cHa, Scer\GAL4^wor.PA, Scer\GAL80^ase.PN, Usp5^leon-1 has type II neuroblast | increased number phenotype, non-suppressible by Usp5^{ED.UAS.Tag:FLAG}, Scer\GAL4^wor.PA, Scer\GAL80^ase.PN

(Komori et al., 2025)

Enhancer of

Statement

Reference

Usp5^leon-2/Usp5^leon-1 is an enhancer of type II neuroblast | increased number phenotype of brat^DG19310/brat¹¹

(Komori et al., 2025)

Usp5^leon-1/Usp5^leon-1 is an enhancer of type II neuroblast | increased number phenotype of brat^DG19310/brat¹¹

(Komori et al., 2025)

Other

Statement

Reference

BacA\p35^UAS.cHa, Scer\GAL4^wor.PA, Scer\GAL80^ase.PN, Usp5^leon-1 has type II neuroblast | increased number phenotype

(Komori et al., 2025)

Additional Comments

Genetic Interactions

Statement

Reference

Xenogenetic Interactions

Statement

Reference

Complementation and Rescue Data

Rescued by

Usp5^19-2/Usp5^leon-1 is rescued by Usp5^GFP

(Wang et al., 2017)

Usp5^19-2/Usp5^leon-1 is rescued by Scer\GAL4^how-24B/Usp5^UAS.Tag:FLAG

(Wang et al., 2017)

Usp5^leon-1 is rescued by Usp5^GFP

(Wang et al., 2014)

Usp5^19-2/Usp5^leon-1 is rescued by Scer\GAL4^Tub.PU/Usp5^UAS.Tag:FLAG

(Wang et al., 2014)

Not rescued by

Usp5^19-2/Usp5^leon-1 is not rescued by Usp5^UAS.Tag:FLAG/Scer\GAL4^Toll-6-D42

(Wang et al., 2017)

Usp5^19-2/Usp5^leon-1 is not rescued by Scer\GAL4^how-24B/Usp5^{ED.UAS.Tag:FLAG}

(Wang et al., 2017)

Usp5^19-2/Usp5^leon-1 is not rescued by Scer\GAL4^Toll-6-D42/Usp5^{ED.UAS.Tag:FLAG}

(Wang et al., 2017)

Usp5^19-2/Usp5^leon-1 is not rescued by Scer\GAL4^Tub.PU/Usp5^{ED.UAS.Tag:FLAG}

(Wang et al., 2014)

Comments

The neuromuscular junction morphological defects (decreased total branch length, reduced number of boutons) observed in Usp5^leon-1/Usp5^19-2 mutant third instar larvae are fully rescued by combination with Usp5^T:Avic\GFP or by expression of Usp5^{Scer\UAS.T:Zzzz\FLAG} driven by Scer\GAL4^how-24B but not if driven by Scer\GAL4^Toll-6-D42, expression of Usp5^{ED.Scer\UAS.T:Zzzz\FLAG} fails to rescue the morphology regardless of the driver.

Usp5^T:Avic\GFP and Usp5^{Scer\UAS.T:Zzzz\FLAG} expression (with the Scer\GAL4^how-24B driver and not with Scer\GAL4^Toll-6-D42) also rescues the impaired electrophysiological properties at Usp5^19-2 or Usp5^leon-1/Usp5^19-2 mutant neuromuscular junctions (reduced excitatory junction current amplitude and quantal content).

(Wang et al., 2017)

Images (0)

Mutant

Wild-type

Stocks (0)

Notes on Origin

Discoverer

External Crossreferences and Linkouts ( 0 )

Synonyms and Secondary IDs (4)

Reported As

Symbol Synonym

Usp5¹

Usp5^leon-1

leon¹

(Pai et al., 2023, Wang et al., 2017, Wang et al., 2014)

usp5^leon1

(Komori et al., 2025)

Name Synonyms

Secondary FlyBase IDs

References (4)

Report Sections

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