Sply05091/Df(2R)BSC433 mutants display reduced viability and decreased foot process density in third instar larval garland cell nephrocytes accompanied by defects in the nephrocyte function.
Sply05091 homozygous mutant adults present a small decrease in survival to starvation, but similar body weight, similar adiposity in abdominal sections, and similar sizes of fat body cells and lipid droplets, as compared to controls.
Sply05091 homozygous mutant adults present decreased feeding behavior, as assessed by decreased starvation-induced appetite, decreased postprandial meal volume, decreased average meal frequency and volume, and decreased daily food intake, as compared to controls.
Heterozygotes are indistinguishable from wild type. Homozygotes show 66.5% lethality. 46% lethality, compared with 3% in wild type, is seen during larval stages, with most larval death occurring during the first instar. 22% lethality, compared with 1% in wild type, is seen during metamorphosis. There is no difference in survival between wild-type and homozygous animals during embryogenesis. Mutant embryos show a pronounced enhancement of apoptosis compared with wild-type controls, especially in a specific region of the posterior pole near the developing genital disc. Muscles appear to develop normally in homozygous embryos and larvae. The dorsal oblique muscles (which serve as templates for the adult dorsal longitudinal muscles) appear normal in homozygous larvae. Homozygous females lay an average of 15.8 +/- 2.98 eggs per day, compared to 44.5 +/- 3.28 per day for wild-type females. The number of eggs produced is significantly reduced in crosses of both male and female mutant flies to wild-type mates. 91% of homozygous adults are flightless, scoring zero in a standard flight performance assay (the average flight score is 0.40 +/- 0.036 compared to 2.60 +/- 0.032 in wild-type flies). Homozygous flies show an accumulation of phosphorylated and unphosphorylated sphingolipids. Homozygous adults show a general pattern of missing dorsal longitudinal flight muscle (DLM) fibres, asymmetry and hypertrophy of remaining fibres. Muscle insertions are not affected. The average number of DLMs per hemithorax is 4.15 +/- 0.21 in the mutant flies, compared to 6.00 +/- 0.00 in wild-type flies. The persistent DLMs have intact myofibrils and normal mitochondria and nuclei. The function of the jump and leg muscles does not appear to be affected in homozygous flies. The giant fiber neuromuscular pathway appears functionally intact by electrophysiology analysis.
Sply05091 has abnormal starvation stress response | adult stage phenotype, enhanceable by lace2/lacek05305
Sply05091 has increased cell death phenotype, suppressible by lace[+]/lacek05305
Sply05091 has lethal | recessive phenotype, suppressible by lace[+]/lacek05305
Sply05091 has flightless | recessive phenotype, suppressible by lace[+]/lacek05305
Sply05091 has semi-sterile | recessive phenotype, suppressible by lace[+]/lacek05305
Sply05091 has abnormal feeding behavior | adult stage phenotype, non-suppressible by lace2/lacek05305
Sply05091/Sply05091 is an enhancer of abnormal starvation stress response | adult stage phenotype of lace2/lacek05305
Sply05091/Sply05091 is a suppressor of lethal | recessive phenotype of lacek05305
Sply05091 has dorsal longitudinal indirect flight muscle cell phenotype, suppressible by lace[+]/lacek05305
Sply05091/Sply05091 is a non-enhancer of adult fat body phenotype of lace2/lacek05305
Sply05091/Sply05091 is a non-enhancer of lipid droplet | adult stage phenotype of lace2/lacek05305
Sply05091/Sply[+] is a suppressor of indirect flight muscle cell phenotype of DysRNAi.C.UAS, Scer\GAL4Tub.PU
Sply05091/Sply[+] is a suppressor of myofibril phenotype of DysRNAi.C.UAS, Scer\GAL4Tub.PU
Sply05091/Sply05091 is a suppressor of wing phenotype of lacek05305
Sply05091/Sply05091 is a suppressor of chaeta phenotype of lacek05305
Sply05091/Sply05091 is a suppressor of eye phenotype of lacek05305
Sply05091/Sply05091 is a suppressor of ommatidium phenotype of lacek05305
One copy of Sply05091 suppresses the defects in indirect flight muscle myofibril integrity seen when DysdsRNA.C.Scer\UAS is expressed under the control of Scer\GAL4tub.PU. This suppression is further enhanced when the flies are given food supplemented with 2-acetyl-4(5)-tetrahydroxybutyl imidazole (THI).
One copy of Sply05091 suppresses the defects in indirect flight muscle myofibril integrity seen in Df(3R)Exel6184 mutant flies. The gross defects in indirect flight muscle morphology are also suppressed.
lacek05305/lace2, Sply05091/Sply05091 adults exhibit a more severe decrease in survival in response to starvation as compared to either lacek05305/lace2 or Sply05091/Sply05091 mutants.
The decreased body weight, decreased adiposity in abdominal sections, and decreased sizes of fat body cells and lipid droplets, observed in lacek05305/lace2 transheterozygous mutant adults is not altered by Sply05091 homozygosity.
The decreased feeding behavior observed in Sply05091 homozygous mutant adults, as assessed by their decreased starvation-induced appetite, decreased postprandial meal volume, decreased average meal frequency and volume, and decreased daily food intake, is not suppressed by lacek05305/lace2 transheterozygosity.
Sply05091/Sply05091 lacek05305/+ adults have an average flight score of 1.41 +/- 0.063 compared to 2.60 +/- 0.032 in wild-type flies in a standard flight performance assay. The average number of dorsal longitudinal flight muscles per hemithorax is 5.13 +/- 0.26 in the mutant flies, compared to 6.00 +/- 0.00 in wild-type flies. Sply05091 lacek05305 double homozygotes show 61% lethality. The eye, bristle and wing defects of lacek05305 surviving adults are suppressed in Sply05091 lacek05305 double homozygotes.
Df(2R)BSC433/Sply05091 is rescued by SplyWT.Tag:HA
Df(2R)BSC433/Sply05091 is rescued by SplyE119G.Tag:HA
Df(2R)BSC433/Sply05091 is not rescued by SplyR210Q.Tag:HA
Df(2R)BSC433/Sply05091 is not rescued by SplyS335I.Tag:HA
The reduced density of foot process in garland cell nephrocytes observed in Sply05091/Df(2R)BSC433 third instar larvae can be rescued by combination with either SplyWT.T:Ivir\HA1 or SplyE119G.T:Ivir\HA1 but not SplyR210Q.T:Ivir\HA1 or SplyS335I.T:Ivir\HA1.
A. Spradling.
Complements: GstS104227. Complements: RhoGEF204291. Complements: l(2)0625306253. Complements: l(2)0665506655. Complements: GstS1k08805. Complements: l(2)k09303k09303. Complements: l(2)k09854k09854. Complements: l(2)k11301k11301. Complements: l(2)k11405k11405.