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Drosophila α-actinin mutant flies exhibit flight muscle paralysis [33], and indeed, upon close inspection using electron microscopy, mutant muscles show irregularities and dissolution of Z-bands, indicating a role for α-actinin in anchoring and stabilizing the thin filaments of the muscles.
Furthermore, mutant flies exhibit abnormalities characteristic of ZS patients, including developmental delay, poor feeding, severe structural abnormalities in the peripheral and central nervous systems, and early death.
Lerp mutant flies exhibit a 30 40% decrease in the level of several lysosomal hydrolases, and are hypersensitive to dietary chloroquine and starvation, consistent with impaired lysosome function.
AKHR 1 ;bmm 1 double mutant flies exhibit an 'obese' phenotype because the fat bodies of these flies store lipids but are not subsequently able to mobilize or release lipids (Grönke et al., 2007).
Although viable and fertile, both homozygous and hemizygous adult mutant flies exhibit a small but statistically significant decrease of ca. 8% (P<0.001 and P<0.0001, respectively) in body mass relative to their respective genetic controls (Fig. 3D).
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Thus the "morning" activity of these double mutant flies exhibits little evidence of gating.
The mutant flies exhibited abnormalities in heavy exercises such as climbing or flight but not in light movements such as locomotion.
ref(2 P mutant flies exhibited several pathological and functional phenotypes reminiscent of those observed in the pink1 or parkin mutants.
The Ir8a and Ir25a double mutant flies exhibited normal post-stimulus aggregation, showing that most ionotropic receptors are not required for the pheromone attraction.
Since LERP is an ortholog of the CI-MPR and has been reported to partially rescue sorting of lysosomal hydrolases in MPR-deficient mammalian cells (Dennes et al., 2005), we were especially interested in determining whether the LERP mutant flies exhibited defects in lysosome biogenesis and function.
show that isoform-mutant flies exhibit defects in male courtship, copulation success, and song production.
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