FlyBase Allele Report: Dmel\Csp[R1]

General Information

Symbol

Dmel\Csp^R1

Species

D. melanogaster

Name

FlyBase ID

FBal0033568

Feature type

allele

Associated gene

Dmel\Csp

Associated Insertion(s)

Carried in Construct

Key Links

Allele class

heat sensitive loss of function allele

loss of function allele

Mutagen

Nature of the Allele

Allele class

heat sensitive loss of function allele

(Umbach et al., 1994, Zinsmaier et al., 1994)

loss of function allele

(Heckmann et al., 1997)

Mutagen

Progenitor genotype

Srpk79D^P1

(Eberle et al., 1998)

Cytology

Description

Imprecise excision of the P{lacW} element, removing the entire Csp gene.

(Eberle et al., 1998)

Deletion of the entire Csp transcription unit from map position -970 to +7800.

(Zinsmaier et al., 1994)

Mutations Mapped to the Genome

Curation Data

Type

Location

Additional Notes

References

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 ( 1 )

Disease

Interaction

References

exacerbates Parkinson's disease 1

modeled by Hsap\SNCA^UAS.cJa

(Yu et al., 2023)

Comments on Models/Modifiers Based on Experimental Evidence ( 0 )

Disease-implicated variant(s)

Phenotypic Data

Phenotypic Class

Phenotype Manifest In

neuromuscular junction | larval stage

(Nie et al., 1999)

NMJ bouton

(Bronk et al., 2005)

Detailed Description

Statement

Reference

Csp^R1 mutants exhibit a reduced number of boutons at neuromuscular junctions (approximately 62% of control).

Evoked EJP amplitudes in Csp^R1 mutant neuromuscular junctions are approximately 52% of wild-type control amplitudes at 22[o]C.

Scer\GAL4^elav-C155-driven expression of Csp^ΔJ.Scer\UAS fails to suppress the temperature-sensitive lethality of Csp^R1 mutants at 25[o]C. Indeed, expression actually enhances the temperature-sensitive lethality of Csp^R1 mutants. Whereas Csp^R1 mutants are fully viable at 18[o]C, only 11% of Csp^R1 mutants expressing Csp^ΔJ.Scer\UAS survive till adulthood.

Scer\GAL4^elav-C155-driven expression of Csp^ΔL.Scer\UAS fails to suppress the temperature-sensitive lethality of Csp^R1 mutants at 25[o]C. Indeed, expression actually enhances the temperature-sensitive lethality of Csp^R1 mutants. Whereas Csp^R1 mutants are fully viable at 18[o]C, only 6% of Csp^R1 mutants expressing Csp^ΔL.Scer\UAS survive till adulthood.

Presynaptic expression of Csp^Scer\UAS.cBa, under the control of Scer\GAL4^elav-C155 in Csp^R1 mutants rescues the loss of synaptic boutons and indeed dramatically increases the number to above wild-type levels (to approximately 186% of wild-type levels).

Expression of Csp^ΔJ.Scer\UAS in motor neurons under the control of Scer\GAL4^D42 increases EJP amplitudes at Csp^R1 mutant neuromuscular junctions to 74% of control amplitudes at 22[o]C. However, normal function is not restored as the amplitudes of EJPs resulting from Csp^ΔJ.Scer\UAS expression are significantly different to wild-type controls.

Csp^R1 mutants exhibit thermo-intolerance of evoked release above 30[o]C, which causes an almost complete loss of evoked release compared with the ~50% reduction of evoked release at 22[o]C.

Increasing the temperature from 22[o]C to 32[o]C in Csp^R1 mutants reduces the average EJP amplitude by approximately 94% (compared to approximately 7% in wild-type controls). No difference is observed in spontaneous quantal release compared to controls. Neither mEJP frequency nor mEJP amplitude are significantly different between 22[o]C and 32[o]C in Csp^R1 mutants.

Csp^R1 mutant boutons exhibit an average resting '[Ca[2+]'] that is significantly increased from 39.9nM at 20[o]C to 145.7nM at 30[o]C, whereas in wild-type controls the average resting '[Ca[2+]'] is unchanged.

Statistically, Ca[2+] concentration levels in Csp^R1 mutant 1[[b]] boutons are not significantly increased from wild-type controls at 20[o]C during and after stimulation. At 30[o]C, however, intraterminal Ca[2+] concentration levels are significantly increased during stimulation in Csp^R1 mutant boutons compared to wild-type.

(Bronk et al., 2005)

The mean evoked excitatory junction potential amplitude at the neuromuscular junction is reduced by about 50% compared to wild type in homozygous larvae. The crumpled wing and eye phenotypes of adult escapers carrying Csp^3.Scer\UAS expressed under the control of Scer\GAL4^elav-C155 are suppressed if the flies are carrying Csp^R1.

(Nie et al., 1999)

Excitatory postsynaptic currents (EPSCs) recorded at 16-18^oC the amplitude is reduced and the time constant of the exponential fit of the current delay is increased. The number of quanta released per pulse does not differ from wild type but the time course of release is distributed differently.

(Heckmann et al., 1997)

At room temperature mutant larvae have sluggish movement. Nerve evoked excitatory junction potentials (EJPs) of larvae are reduced to half the level of wild type. Mutation does not affect the number of calcium binding sites needed to release a single quantum so there must have been a change in the number or properties of the calcium channel/transmitter release assemblies that led to a reduction of quantal output in response to nerve stimulation. Action potential propagation and postsynaptic events in receptor function are unaffected at high temperatures, so cannot explain the temperature- dependent loss of EJPs. Evoked neuromuscular transmission is blocked at high temperatures (30^oC), the block is reversible when the temperature is reduced from 30^oC to 22^oC.

(Umbach et al., 1994)

At 29^oC flies become paralysed and die. Semilethal embryonic phenotype, adult escapers die within 4-5 days, there are no adult escapers at 29^oC.

(Zinsmaier et al., 1994)

External Data

Linkouts

Interactions

Show genetic interaction network for Enhancers & Suppressors

Phenotypic Class

Enhancer of

Statement

Reference

Csp^R1 is an enhancer of abnormal neurophysiology | adult stage phenotype of Hsap\MAPT^{P301L.QUAS.0N4R}, Ncra\QF^QF2w.nSyb

(Praschberger et al., 2023)

Phenotype Manifest In

Enhancer of

Statement

Reference

Csp^R1 is an enhancer of retina | adult stage phenotype of Hsap\MAPT^{P301L.QUAS.0N4R}, Ncra\QF^QF2w.nSyb

(Praschberger et al., 2023)

Additional Comments

Genetic Interactions

Statement

Reference

Xenogenetic Interactions

Statement

Reference

Complementation and Rescue Data

Fails to complement

Csp^X1

(Eberle et al., 1998)

Rescued by

Csp^R1 is rescued by Scer\GAL4^elav-C155/Csp^H45Q.UAS

(Bronk et al., 2005)

Csp^R1 is rescued by Scer\GAL4^elav-C155/Csp^ΔJ.UAS

(Bronk et al., 2005)

Csp^R1 is rescued by Csp^ΔJ.UAS/Scer\GAL4^Toll-6-D42

(Bronk et al., 2005)

Csp^R1 is rescued by Scer\GAL4^Toll-6-D42/Csp^ΔL.UAS

(Bronk et al., 2005)

Csp^R1 is rescued by Scer\GAL4^elav-C155/Csp^ΔL.UAS

(Bronk et al., 2005)

Csp^R1 is rescued by Csp^+t12.3

(Zinsmaier et al., 1994)

Partially rescued by

Csp^R1 is partially rescued by Scer\GAL4^elav-C155/Csp^UAS.cBa

(Bronk et al., 2005)

Csp^R1 is partially rescued by Scer\GAL4^Toll-6-D42/Csp^H45Q.UAS

(Bronk et al., 2005)

Csp^R1 is partially rescued by Scer\GAL4^elav-C155/Csp^2.UAS

(Nie et al., 1999)

Csp^R1 is partially rescued by Scer\GAL4^elav-C155/Csp^1.UAS

(Nie et al., 1999)

Csp^R1 is partially rescued by Scer\GAL4^elav-C155/Csp^3.UAS

(Nie et al., 1999)

Not rescued by

Csp^R1 is not rescued by Scer\GAL4^elav-C155/Csp^ΔJ.UAS

(Bronk et al., 2005)

Csp^R1 is not rescued by Csp^ΔJ.UAS/Scer\GAL4^Toll-6-D42

(Bronk et al., 2005)

Csp^R1 is not rescued by Scer\GAL4^Toll-6-D42/Csp^ΔL.UAS

(Bronk et al., 2005)

Csp^R1 is not rescued by Scer\GAL4^elav-C155/Csp^ΔL.UAS

(Bronk et al., 2005)

Comments

Expression of Csp^ΔJ.Scer\UAS (under the control of Scer\GAL4^D42) restores the thermo-tolerance of evoked release at Csp^R1 mutant neuromuscular junctions. No difference is observed in spontaneous quantal release compared to controls. Neither mEJP frequency nor mEJP amplitude are significantly different between 22[o]C and 32[o]C in Csp^R1 mutants expressing Csp^ΔJ.Scer\UAS (under the control of Scer\GAL4^D42).

Expression of Csp^ΔJ.Scer\UAS at Csp^R1 mutant neuromuscular junctions (under the control of Scer\GAL4^D42) has little effect on the abnormally increased frequency of asynchronous release shortly after the evoked EJP.

Motor neuron expression of Csp^ΔJ.Scer\UAS (under the control of Scer\GAL4^D42) has no significant effect on the elevated intraterminal '[Ca[2+]'] of Csp^R1 mutant neuromuscular junctions.

Expression of Csp^ΔJ.Scer\UAS in motor neurons (under the control of Scer\GAL4^D42) has no significant effect on Ca[2+] levels in Csp^R1 mutant motor terminals before, during, and after stimulation at 20 and 30[o]C.

Presynaptic expression of Csp^ΔJ.Scer\UAS, under the control of Scer\GAL4^elav-C155 in Csp^R1 mutants rescues the loss of synaptic boutons, increasing their number to approximately 74% of wild-type levels.

Expression of Csp^ΔJ.Scer\UAS in Csp^R1 mutant motor terminals (under the control of Scer\GAL4^D42) has no significant effect on '[Ca[2+]'] levels before, during, and after stimulation at 20 and 30[o]C.

Expression of Csp^ΔJ.Scer\UAS at Csp^R1 mutant neuromuscular junctions (under the control of Scer\GAL4^D42), restores the thermo-tolerance of release: at 32[o]C, the average EJP amplitude is slight reduced by 9% but is not significantly different to wild-type controls. Neither mEJP frequency nor mEJP amplitude is significantly different between the groups at 22 or 32[o]C.

Scer\GAL4^elav-C155-driven expression of Csp^ΔJ.Scer\UAS fails to suppress the temperature-sensitive lethality of Csp^R1 mutants at 25[o]C.

Expression of Csp^ΔL.Scer\UAS in motor neurons (under the control of Scer\GAL4^D42) has no significant effect on Ca[2+] levels in Csp^R1 mutant motor terminals before, during, and after stimulation at 20 and 30[o]C.

Expression of Csp^{H45Q.Scer\UAS} in motor neurons under the control of Scer\GAL4^D42 increases EJP amplitudes at Csp^R1 mutant neuromuscular junctions. The mean EJP amplitude in larvae expressing Csp^{H45Q.Scer\UAS} is not significantly different to wild-type controls. Although Csp^{H45Q.Scer\UAS} expression restores ELP amplitudes to control levels, the Csp^{H45Q.Scer\UAS}-mediated effect is not very robust. The coefficient of variation of EJP amplitudes is 0.08 for controls, whereas for Csp^{H45Q.Scer\UAS} it's 0.24, indicating expression of Csp^{H45Q.Scer\UAS} does not fully restore synaptic transmission.

Presynaptic expression of Csp^ΔL.Scer\UAS, under the control of Scer\GAL4^elav-C155 in Csp^R1 mutants rescues the loss of synaptic boutons, increasing their number to approximately 80% of wild-type levels.

Presynaptic expression of Csp^{H45Q.Scer\UAS}, under the control of Scer\GAL4^elav-C155 in Csp^R1 mutants rescues the loss of synaptic boutons, increasing their number to approximately 140% of wild-type levels.

Scer\GAL4^elav-C155-driven expression of Csp^Scer\UAS.cBa partially suppresses (62%) the temperature-sensitive lethality of Csp^R1 mutants at 25[o]C.

Expression of Csp^ΔL.Scer\UAS (under the control of Scer\GAL4^D42) restores the thermo-tolerance of evoked release at Csp^R1 mutant neuromuscular junctions. No difference is observed in spontaneous quantal release compared to controls. Neither mEJP frequency nor mEJP amplitude are significantly different between 22[o]C and 32[o]C in Csp^R1 mutants expressing Csp^ΔL.Scer\UAS (under the control of Scer\GAL4^D42).

Expression of Csp^ΔL.Scer\UAS at Csp^R1 mutant neuromuscular junctions (under the control of Scer\GAL4^D42) greatly reduces the abnormally increased frequency of asynchronous release shortly after the evoked EJP.

At 30[o]C, motor neuron-specific expression of Csp^ΔL.Scer\UAS (under the control of Scer\GAL4^D42 partially reverses the abnormally high '[Ca[2+]'] resting levels of Csp^R1 mutant boutons to an intermediate level of 93.4nM. Statistically, '[Ca[2+]'] levels at 30[o]C are significantly different from those seen in Csp^R1 mutants and Csp^R1 mutants expressing Csp^ΔJ.Scer\UAS. At 20[o]C, expression of Csp^ΔL.Scer\UAS has no significant effect on the intraterminal '[Ca[2+]'].

Expression of Csp^ΔL.Scer\UAS in Csp^R1 mutant neuromuscular junctions (under the control of Scer\GAL4^D42) partially restores normal intraterminal

'[Ca[2+]'] levels at 30[o]C. Ca[2+] signals at rest, during and after stimulation, reaches intermediate levels, being significantly higher than those of wild-type controls at 30[o]C, but also clearly lower than those of Csp^R1 mutants.

Expression of Csp^ΔL.Scer\UAS at Csp^R1 mutant neuromuscular junctions (under the control of Scer\GAL4^D42), restores the thermo-tolerance of release: at 32[o]C, the average EJP amplitude is slight reduced by 16% but is not significantly different to wild-type controls. Neither mEJP frequency onor mEJP amplitude is significantly different between the groups at 22 or 32[o]C.

Expression of Csp^ΔL.Scer\UAS at Csp^R1 mutant neuromuscular junctions (under the control of Scer\GAL4^D42) significantly reduces the frequency of asynchronous release shortly after the evoked EJP.

(Bronk et al., 2005)

Expression of Csp^1.Scer\UAS, Csp^2.Scer\UAS or Csp^3.Scer\UAS under the control of Scer\GAL4^elav-C155 has no effect on spontaneous miniature excitatory junction potentials at the neuromuscular junction of Csp^R1 third instar larvae, but does rescue the evoked excitatory junction potential at the neuromuscular junction.

(Nie et al., 1999)

Semilethality of homozygotes can be rescued by introduction of the P{Csp12.3} construct.

(Zinsmaier et al., 1994)

Images (0)

Mutant

Wild-type

Stocks (1)

Bloomington

51998

w^*; Csp^R1/TM6C, Tb¹ Sb¹

Notes on Origin

Discoverer

Comments

All heteroallelic combinations of Csp are semi-lethal.

(Zinsmaier et al., 1994)

External Crossreferences and Linkouts ( 0 )

Synonyms and Secondary IDs (2)

Reported As

Symbol Synonym

Csp^R1

(Praschberger et al., 2023, Yu et al., 2023, Wong et al., 2005)

csp^R1

(Imler et al., 2019, Bronk et al., 2005, Nie et al., 1999, Eberle et al., 1998, Heckmann et al., 1997, Umbach et al., 1994, Zinsmaier et al., 1994)

Name Synonyms

Secondary FlyBase IDs

References (10)

Report Sections

Open Close