Imprecise excision of P{EP}cazEP1564 resulting in a deletion that removes 2443 bp of the caz coding sequence and 3251 bp of the upstream region including the entire caz promoter. The deletion also disrupts CG32576.
Approximate boundaries of the caz383 deletion, which was reported as a small deletion that removes 2,443 bp of the caz gene and 3,251 bp of upstream sequences including the entire caz promoter. The boundaries are approximate because it's not clear what coordinate was used as the beginning of the caz gene. The deletion was mapped from a transcription start at X:16182879 (R5 coordinates).
visible, with CG32576+t5.0
caz383 homozygotes are semi-lethal and the few eclosing adults are not able to climb. These mutants also show impaired larval locomotion in crawling assays, as compared to controls.
caz383 homozygotes show a decrease in the axonal transport both of mitochondria and of (NPY-positive) vesicles (i.e. significant increases in stationary fraction) in third instar larval motor neurons, as compared to controls.
Third instar larvae which are mutant for 'caz1' (FlyBase curator comment: this mutant corresponds to Df(1)383 onto which a transgene containing CG32576+t5.0 has been recombined to provide CG32576[+] function) display increased evoked excitatory junction potential (EJP) amplitude compared with controls. This increase is due to an approximately 25% increase in mEJP amplitude without affecting quantal content. The mutants have normal mEJP frequency. Active zones of caz1 mutant animals appear normal.
The neuromuscular junctions of 'caz1' mutants (FlyBase curator comment: this mutant corresponds to Df(1)383 onto which a transgene containing CG32576+t5.0 has been recombined to provide CG32576[+] function) experience a decrease in the amplitude of excitatory junctional currents (EJCs), compared to wild-type. The rate of EJC decay in markedly increased in caz1 mutants compared to controls (decay time constants are approximately 35% of those in controls). This substantial change in decay kinetics, in combination with the decrease in EJC amplitude, results in an even more profound decrease in the integrated synaptic current.
Male larvae hemizygous for the 'caz1' mutant chromosome (Df(1)383 onto which a transgene containing CG32576+t5.0 has been recombined to provide CG32576[+] function) are morphologically normal. However, only 14% of these animals eclose as adults.
caz1/Y adult escapers show a 57% decrease in average life span compared to controls. They also show a number of external defects, including a mild rough eye phenotype, abnormal genitalia and defects in both bristles and wing vein organisation.
caz1/Y adult escapers have prominent locomotion defects. Most are unable to fly, and compared to controls, they walk slowly, often fall over, and have difficulty righting themselves. They show a 55.9% decrease in climbing ability in a negative geotaxis assay abd a 67.6% decrease in locomotor speed compared to controls.
The brains of 25 day old caz1/Y adult escapers do not show evidence of vacuolization or other evidence of extensive neuronal death.
caz383 has partially lethal - majority die phenotype, suppressible by Hsap\FUSUAS.10x.Tag:FLAG/Scer\GAL4Toll-6-D42
caz383 has abnormal locomotor behavior | adult stage phenotype, suppressible | partially by Hsap\FUSUAS.10x.Tag:FLAG/Scer\GAL4Toll-6-D42
caz383 has abnormal locomotor behavior | third instar larval stage phenotype, suppressible | partially by Hsap\FUSUAS.10x.Tag:FLAG/Scer\GAL4Toll-6-D42
caz383 has partially lethal - majority die phenotype, suppressible | partially by Hsap\FUSP525L.10x.UAS.Tag:FLAG/Scer\GAL4Toll-6-D42
CG32576+t5.0, caz383 has short lived phenotype, suppressible | partially by Scer\GAL4elav-C155/Hsap\FUSP525L.10x.UAS.Tag:FLAG
CG32576+t5.0, caz383 has short lived phenotype, suppressible | partially by Scer\GAL4elav-C155/Hsap\FUSR522G.10x.UAS.Tag:FLAG
CG32576+t5.0, caz383 has partially lethal - majority die phenotype, suppressible by Scer\GAL4elav-C155/Hsap\FUSUAS.10x.Tag:FLAG
CG32576+t5.0, caz383 has partially lethal - majority die phenotype, suppressible by Scer\GAL4elav-C155/Hsap\FUSP525L.10x.UAS.Tag:FLAG
CG32576+t5.0, caz383 has partially lethal - majority die phenotype, suppressible by Scer\GAL4elav-C155/Hsap\FUSR522G.10x.UAS.Tag:FLAG
CG32576+t5.0, caz383 has abnormal locomotor behavior phenotype, suppressible | partially by Scer\GAL4elav-C155/Hsap\FUSUAS.10x.Tag:FLAG
CG32576+t5.0, caz383 has abnormal locomotor behavior phenotype, suppressible | partially by Scer\GAL4elav-C155/Hsap\FUSP525L.10x.UAS.Tag:FLAG
CG32576+t5.0, caz383 has short lived phenotype, suppressible by Scer\GAL4elav-C155/Hsap\FUSUAS.10x.Tag:FLAG
CG32576+t5.0, caz383 has partially lethal - majority die phenotype, non-suppressible by Scer\GAL4elav-C155/TBPHUAS.10x.Venus
CG32576+t5.0, caz383 has short lived phenotype, non-suppressible by Scer\GAL4elav-C155/TBPHUAS.10x.Venus
CG32576+t5.0, caz383 has abnormal locomotor behavior phenotype, non-suppressible by Scer\GAL4elav-C155/TBPHUAS.10x.Venus
CG32576+t5.0, caz383 has abnormal locomotor behavior phenotype, non-suppressible by Scer\GAL4elav-C155/Hsap\FUSR522G.10x.UAS.Tag:FLAG
caz383 has embryonic/larval motor neuron | third instar larval stage phenotype, suppressible by Scer\GAL4CCAP.PU/Hsap\FUSUAS.10x.Tag:FLAG
caz383 has embryonic/larval motor neuron | third instar larval stage phenotype, suppressible | partially by Scer\GAL4CCAP.PU/Hsap\FUSP525L.10x.UAS.Tag:FLAG
Expression of TBPHScer\UAS.10x.T:Avic\GFP-YFP.Venus in the nervous system under the control of Scer\GAL4elav-C155 does not rescue the reduced viability, locomotion defects or reduced longevity of adult 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 neuromuscular junction expansion induced by the overexpression of TBPHScer\UAS.10x.T:Avic\GFP-YFP.Venus under the control of Scer\GAL4RapGAP1-OK6 is completely suppressed in a 'caz1'/Y background.
The decreased axonal transport of mitochondria in caz383 homozygous third instar larval motor neurons is rescued by the expression of either Hsap\FUSScer\UAS.10x.T:Zzzz\FLAG or Hsap\FUSP525L.10x.Scer\UAS.T:Zzzz\FLAG under the control of Scer\GAL4CCAP.PI; their decreased axonal transport of (NPY-positive) vesicles is rescued by the expression of Hsap\FUSScer\UAS.10x.T:Zzzz\FLAG, but not or Hsap\FUSP525L.10x.Scer\UAS.T:Zzzz\FLAG, under the control of Scer\GAL4CCAP.PI.
The semi-lethality, the decreased third instar larval crawling capacity and the decreased adult climbing capacity observed in caz383 homozygotes are fully/nearly fully suppressed by the expression of either Hsap\FUSScer\UAS.10x.T:Zzzz\FLAG or Hsap\FUSP525L.10x.Scer\UAS.T:Zzzz\FLAG under the control of Scer\GAL4Toll-6-D42.
Expression of either Hsap\FUSScer\UAS.10x.T:Zzzz\FLAG, Hsap\FUSP525L.10x.Scer\UAS.T:Zzzz\FLAG or Hsap\FUSR522G.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 each Hsap\FUS transgene is not significantly different from that seen when a transgene containing wild-type caz is expressed using the same driver.
Expression of Hsap\FUSScer\UAS.10x.T:Zzzz\FLAG under the control of Scer\GAL4elav-C155 strongly rescues the locomotor defects seen in caz1/Y adult males, to a level that 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\FUSR522G.10x.Scer\UAS.T:Zzzz\FLAG under the control of Scer\GAL4elav-C155 does not rescue the locomotor defects seen in caz1/Y adult males.
Expression of Hsap\FUSScer\UAS.10x.T:Zzzz\FLAG under the control of Scer\GAL4elav-C155 completely rescues the reduced longevity of adult caz1/Y escapers.
Expression of either Hsap\FUSP525L.10x.Scer\UAS.T:Zzzz\FLAG or Hsap\FUSR522G.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.
caz383 is rescued by Scer\GAL4CCAP.PU/cazUAS.x10.Tag:FLAG
caz383 is rescued by cazUAS.x10.Tag:FLAG/Scer\GAL4Toll-6-D42
caz383 is rescued by Scer\GAL4Toll-6-D42/cazP398L.10x.UAS.Tag:FLAG
caz383 is partially rescued by Scer\GAL4CCAP.PU/cazP398L.10x.UAS.Tag:FLAG
The semi-lethality, the decreased third instar larval crawling capacity and the decreased adult climbing capacity observed in caz383 homozygotes are rescued by the expression of either cazScer\UAS.x10.T:Zzzz\FLAG or cazP398L.10x.Scer\UAS.T:Zzzz\FLAG under the control of Scer\GAL4Toll-6-D42.
The decreased axonal transport of mitochondria in caz383 homozygous third instar larval motor neurons is rescued by the expression of either cazScer\UAS.x10.T:Zzzz\FLAG or cazP398L.10x.Scer\UAS.T:Zzzz\FLAG under the control of Scer\GAL4CCAP.PI; their decreased axonal transport of (NPY-positive) vesicles is rescued by the expression of cazScer\UAS.x10.T:Zzzz\FLAG, but not or cazP398L.10x.Scer\UAS.T:Zzzz\FLAG, under the control of Scer\GAL4CCAP.PI.
cazT:Zzzz\FLAG rescues the electrophysiological defects seen in 'caz1' mutants (FlyBase curator comment: this mutant corresponds to Df(1)383 onto which a transgene containing CG32576+t5.0 has been recombined to provide CG32576[+] function).
caz+tBID rescues the lethality of males hemizygous for the 'caz1' chromosome (FlyBase curator comment: this chromosome corresponds to Df(1)383 onto which a transgene containing CG32576+t5.0 has been recombined to provide CG32576[+] function).
cazT:Zzzz\FLAG fully rescues the reduced 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 reduced longevity, locomotion defects and external morphological defects of adult caz1/Y escapers are also fully rescued by cazT:Zzzz\FLAG.
Expression of cazScer\UAS.x10.T:Zzzz\FLAG in the nervous system under the control of Scer\GAL4elav-C155 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) to 83.7% of control levels. The locomotion defects seen in caz1/Y adult escapers are also strongly, but not completely rescued in these animals, while longevity is completely rescued.