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General Information
Symbol
Dmel\Fmr1UAS.cZa
Species
D. melanogaster
Name
Saccharomyces cerevisiae UAS construct a of Zhang
FlyBase ID
FBal0131027
Feature type
allele
Associated gene
Associated Insertion(s)
Carried in Construct
Also Known As
UAS-dfmr1, UAS-9557-3, UAS-dfmr, UAS-dfxr, UAS-dFMRP, UAS-dfmr19557-3
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

Expression of Fmr1 is governed by UASt regulatory sequences.

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

gamma-lobe & neuron | somatic clone, with Scer\GAL4ey-OK107

mushroom body & neuron & dendrite | somatic clone, with Scer\GAL4ey-OK107

mushroom body & neuron | somatic clone, with Scer\GAL4ey-OK107

photoreceptor cell & synapse & lamina receptor cell, with Scer\GAL4hs.2sev

Detailed Description
Statement
Reference

Third instar larvae expressing Fmr1UAS.cZa under the control of Scer\GAL4elav-C155 have significantly less mature and satellite NMJ boutons than controls.

Expressing Fmr1UAS.cZa under the control of Scer\GAL4elav.PU does not significantly affect the third instar larval neuromuscular junction, compared to controls.

Expression of Fmr1Scer\UAS.cZa under the control of Scer\GAL4ptc-559.1 results in reduction in the size of the wing region between wing veins 3 and 4, and in some cases, modest degradation of the basement membrane in the hinge region of wing imaginal discs.

Expression of Fmr1Scer\UAS.cZa under the control of Scer\GAL4vg.PM results in wing notches.

Targeted, constitutive Fmr1 expression within the sLN[[v]] neurons, through expression of Fmr1Scer\UAS.cZa under the control of Scer\GAL4P2.4.Pdf, results in the collapse and retraction of the terminal synaptic arbor, evident in the reduction of synaptic area, loss of synaptic varicosities and a complex lack of outward projecting axonal branches.

Overexpression of Fmr1Scer\UAS.cZa at the earliest stage of pupal development, under the control of RU486-induced Scer\GAL4elav.Switch.PO exacerbates the Fmr1Δ50M mutant phenotype resulting in an elevated number of Pdf-positive synaptic boutons redistributed within 30% of the sLN[[v]] axonal arbor. Similarly, induction at pupal stage P2 provides no significant influence upon the synaptic arborization, with no change in the total number of Pdf-positive boutons.

Overexpression of Fmr1Scer\UAS.cZa in the latter stage of pupal development, under the control of RU486-induced Scer\GAL4elav.Switch.PO ameliorates the Fmr1Δ50M mutant phenotype, restricting the number and specificity of synaptic connections.

Acute, adult, heat shock-induced expression of Fmr1Scer\UAS.cZa produces a significant decrease in 1-d memory after spaced training but overexpression of Fmr1Scer\UAS.cZa after spaced training beyond memory consolidation does not affect 1-d memory. Massed training remains unaffected by any overexpression procedure. Learning appears to be normal at 3 or 24 hours after overexpression in adult flies.

The clonal expression of Fmr1UAS.cZa (under the control of Scer\GAL4ey-OK107) in gamma neurons leads to fewer and shorter branches at prepupal stage 3, and abrogates the expected axonal growth, but does not affect branch number or length, at prepupal stage 4.

Flies that express Fmr1Scer\UAS.cZa, under the control of Scer\GAL4P2.4.Pdf, almost completely lack wild-type target innervation by the LNv. In the brains of flies that overexpress Fmr1Scer\UAS.cZa under the control of Scer\GAL4c747, there is a significant increase in the gap between the β lobes and between the γ lobes of the mushroom body.

Expression of Fmr1Scer\UAS.cZa under the control of Scer\GAL4OK107 results in dramatic defects in the mushroom body axon lobes; the β lobe always overextends across the midline, the β' lobe often shows apparently random directions of axon projection which are often outside of the mushroom body domain and the γ lobe always shows a dramatic decrease in volume and is sometimes completely lost. The number of cell body processes produced by neuronal cells in the mushroom body is reduced 2-fold in single cell clones expressing Fmr1Scer\UAS.cZa under the control of Scer\GAL4OK107 compared to control single cell clones. The "claw-like" structure normally seen at the terminal of dendritic branches (this consists of a fine dendritic terminal arbor) is severely reduced or completely absent in the mutant cells. Single γ neurons in the mushroom body that express Fmr1Scer\UAS.cZa under the control of Scer\GAL4OK107 in an otherwise wild-type background show underbranching and dramatically reduced growth. Most of these mutant γ neurons maintain their process along the ventral edge of the horizontal lobe and aberrantly invade the β lobe.

When expression is driven by Scer\GAL4ato.3.6 axon extension from the lobula to the medulla fails completely. Fibers enter the lobula but then either project to inappropriate sites within the lobula or stop at the lobula-medulla border.

When expression is driven by Scer\GAL4elav-C155 flies show abnormally outspread wings and cannot fly. Flies are uncoordinated and show adult lethality - dying 5-10 days post-eclosion. When expression is driven by Scer\GAL4Mhc.PW flies' wings are droopy or held up and they cannot fly. When expression is driven by Scer\GAL4G7 flies show pupal lethality. When expression is driven by Scer\GAL4hs.2sev the retina is disordered with misshapen rhabdomeres, abnormal rhabdomere numbers and fused ommatidia. Phototransduction is normal, however synaptic transmission is reduced (as indicated by a reduction in off-transient mean amplitude as assayed by ERG). When expression is driven by Scer\GAL4elav-C155 the larval neuromuscular junction shows pronounced synaptic undergrowth and underelaboration of synaptic terminals. Muscle 4 has 36% decrease in number of boutons over controls. Bouton diameter is nearly twice that of wild type. When expression is driven by Scer\GAL4Mhc.PW the larval neuromuscular junction shows pronounced synaptic undergrowth and underelaboration of synaptic terminals. Muscle 4 has 17% decrease in number of boutons over controls. Mean EJC amplitude is unaffected when expression is driven by Scer\GAL4elav-C155 but mEJC analysis shows that the frequency of spontaneous glutamate release is increased five-fold (though unaffected when driven by Scer\GAL4Mhc.PW).

External Data
Interactions
Show genetic interaction network for Enhancers & Suppressors
Phenotypic Class
Suppressed by
NOT suppressed by
Suppressor of
Phenotype Manifest In
Suppressed by
NOT suppressed by
Suppressor of
NOT Suppressor of
Statement
Reference
Additional Comments
Genetic Interactions
Statement
Reference

The co-expression of Fmr1Scer\UAS.cZa under the control of Scer\GAL4c135, but not Scer\GAL4nos.PU, suppresses the formation of crystalline aggregates (of Stellate protein) in spermatocytes upon aubGD11831 expression.

The dramatic overgrowth of presynaptic arbors found in hiwΔNterm mutant C4 da neurons is completely abolished by expression of Fmr1Scer\UAS.cZa under the control of Scer\GAL4ppk.PG.

Flies that co-express chicScer\UAS.cCa and Fmr1Scer\UAS.cZa, both under the control of Scer\GAL4P2.4.Pdf, show a wild-type-like pattern of LNv innervation. The LNv fasciculation phenotype seen when chicScer\UAS.cCa is expressed alone is suppressed. Coexpression of chicScer\UAS.cCa significantly reduces the mushroom body phenotype seen when flies overexpress Fmr1Scer\UAS.cZa under the control of Scer\GAL4c747.

Xenogenetic Interactions
Statement
Reference
Complementation and Rescue Data
Partially rescues
Comments

The expression of Fmr1Scer\UAS.cZa under the control of Scer\GAL4T76 rescues the lethality of Fmr1Δ113M homozygotes and suppresses the crystalline aggregates observed in the spermatocytes of Fmr1Δ113M homozygotes and heterozygotes; this expression also partially rescues the male sterility of Fmr1Δ50M homozygotes and of Fmr1Δ50M/Fmr1Δ113M transheterozygotes, and partially rescues the decreased fertilities of Fmr1Δ50M heterozygous males, Fmr1Δ50M homozygous and heterozygous females, and Fmr1Δ50M/Fmr1Δ113M transheterozygous females.

The expression of Fmr1Scer\UAS.cZa under the control of Scer\GAL4c135, Scer\GAL4nos.PU, or Scer\GAL4upd1.PU suppresses the crystalline aggregates observed in the spermatocytes of Fmr1Δ113M heterozygotes.

Expression of Fmr1Scer\UAS.cZa under the control of Scer\GAL4GMR12G04 (either in combination with a Gal80[ts] transgene to restrict expression to the period including pupal day 4 to 1 day post-eclosion, or without a Gal80[ts] for expression throughout development) significantly rescues the MVP2 dendritic arbor overgrowth seen in Fmr1Δ50M/Fmr1Δ50M mutants.

Expression of Fmr1Scer\UAS.cZa under the control of Scer\GAL4GMR65G01 (either in combination with a Gal80[ts] transgene to restrict expression to the period including pupal day 4 to 1 day post-eclosion, or without a Gal80[ts] for expression throughout development) significantly rescues the projection neuron dendritic arbor overgrowth seen in Fmr1Δ50M/Fmr1Δ50M mutants.

Provision of Fmr1 transiently during the early stages of neural development, through expression of Fmr1Scer\UAS.cZa under the RU486-induced control of Scer\GAL4elav.Switch.PO in a Fmr1Δ50M mutant background is not sufficient to correct the characteristic synaptic defects caused by the loss of Fmr1 as assayed at maturity. Provision of Fmr1 at maturity, following completion of neural development, is not sufficient to rescue the synaptic defects found in Fmr1Δ50M mutants.

Images (0)
Mutant
Wild-type
Stocks (1)
Notes on Origin
Discoverer
External Crossreferences and Linkouts ( 0 )
Synonyms and Secondary IDs (4)
Reported As
Symbol Synonym
Fmr1Scer\UAS.cZa
Fmr1UAS.cZa
Name Synonyms
Saccharomyces cerevisiae UAS construct a of Zhang
Secondary FlyBase IDs
    References (25)