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General Information
Symbol
Dmel\Hr51X1
Species
D. melanogaster
Name
FlyBase ID
FBal0239784
Feature type
allele
Associated gene
Associated Insertion(s)
Carried in Construct
Also Known As
unfX1
Key Links
Allele class
Nature of the Allele
Allele class
Mutations Mapped to the Genome
 
Type
Location
Additional Notes
References
Nucleotide change:

G15333968C

Comment:

Splice donor mutation in the first base of intron two. An XbaI site was also introduced. Created by homologous recombination.

Associated Sequence Data
DNA sequence
Protein sequence
 
 
Progenitor genotype
Cytology
Nature of the lesion
Statement
Reference

Hr51X1 carries a mutation in the second exon.

Mutation of the first base of Hr51 intron 2 from a "g" to a "C", which is predicted to destroy the 5' donor splice site of intron 2 and to result in an in-frame stop codon, producing a truncated protein of 145 amino acid residues that lacks the second zinc finger, the hinge region and the ligand-binding domain. In addition, bases 5 and 6 of the endogenous intron 2 sequence have been mutated and 4 bases inserted to produce an XbaI site.

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

Homozygous Hr51X1 mutant flies exhibit a somewhat unusual but diurnal activity pattern in LD conditions (12 hours light: 12 hours dark) that have a delayed morning activity peak and an advanced evening activity peak. The majority of homozygous Hr51X1 flies are arrhythmic in DD conditions (constant darkness). No defects are seen in heterozygotes in either LD or DD.

Hr51X1/Df(2R)ED2426 mutant flies are arrhythmic in both LD conditions (12 hours light: 12 hours dark) and DD (constant darkness).

Hr51X1/Hr51Z0001 mutant flies are arrhythmic in both LD conditions (12 hours light: 12 hours dark) and DD (constant darkness).

Hr51X1 mutant neuroblast clones exhibit γ-lobe defects with a decrease in innervation of the adult γ-lobe. α/β neurons exhibit normal morphology and show no signs of axon stalling.

Hr51X1 heterozygous MARCM clones exhibit guidance defects in α/β neurons.

Hr51X1/Df(2R)ED2426 hemizygous and Hr51X1 homozygous mutants show a complete loss of mushroom body (MB) lobes. MB axons of adult hemizygous and homozygous mutants project anteroventrally, forming a peduncle, but stall prior to the formation of discrete lobes. Axons that reach the heel region of the MB tend to spread out, but do not extend axons medially or dorsally. γ neurons extend axons medially but not dorsally in Hr51X1/Df(2R)ED2426 larvae and they undergo pruning like wild-type axons at approximately 16 hours after puparium formation (APF). However, they fail to re-extend during pupal development. α'/β' and α/β projections are never observed in Hr51X1/Df(2R)ED2426 pupae at 48 hours APF.

Hr51X1/Hr51Z0001 heterozygotes exhibit a range of mushroom body (MB) phenotypes, for example, missing MB lobes.

Hr51Z0001/Df(2R)ED2426 can show reduced eclosion, although the phenotype is variable in different crosses.

Approximately a quarter of Hr51X1/Df(2R)ED2426 adults have unexpanded wings.

44% of homozygous flies fail to eclose and 17% eclose but have unexpanded wings.

15% of Hr51X1/Hr51Z0001 flies fail to eclose and 62% eclose but have unexpanded wings.

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
Fails to complement
Partially rescued by
Comments

Expression of Hr51+tCH322-72C6 partially rescues the locomotor rhythm defects seen in Hr51X1/Df(2R)ED2426 mutants. Free-running rhythms are restored in 80% of flies.

Medial and dorsal mushroom body (MB) lobes are observed in all Hr51X1/Df(2R)ED2426 animals expressing Hr51Scer\UAS.cBa driven by Scer\GAL4ey-OK107, but MB lobes appear thin and less robust in some rescued animals. The medially projecting axons in rescued animals fail to stop appropriately, extending axons past the midline.

Images (0)
Mutant
Wild-type
Stocks (1)
Notes on Origin
Discoverer
External Crossreferences and Linkouts ( 0 )
Synonyms and Secondary IDs (3)
Reported As
Name Synonyms
Secondary FlyBase IDs
    References (5)