Hsap\ARQ52.Scer\UAS disease model requires Hsap\AR ligand testosterone. Larvae raised in the presence of dihydroxytestosterone.
Head eversion occurs at 15 hours after puparium formation in ctpG0371 mutant pupae. 12 hours after head eversion, a defect in salivary gland death is evident. All pupae fail to complete salivary gland degradation, compared to control animals which completely lack salivary glands at this stage. 39% of mutant pupae have intact glands, and 61% display partially fragmented salivary glands.
Autophagy is defective in ctpG0371 mutant salivary glands, and the number of autophagosomes present in salivary gland cells is reduced compared to controls. ctpG0371 mutant salivary gland cells contain protein inclusions.
ctpG0371 mutant third instar larvae display motility defects. ctpG0371 mutants possess decreased neuromuscular junction bouton numbers compared to controls, and protein inclusions are present in larval muscles.
ctpG0371 is an enhancer of abnormal locomotor behavior | conditional phenotype of Hsap\ARQ52.UAS, Scer\GAL4Toll-6-D42
ctpG0371 has salivary gland | pupal stage phenotype, enhanceable by Scer\GAL4da.G32/BacA\p35UAS.cHa
ctpG0371 has salivary gland | pupal stage phenotype, suppressible by Scer\GAL4fkh.PH/Atg1GS10797
Expression of Atg1GS10797 in the salivary glands under the control of Scer\GAL4fkh.PH in ctpG0371 mutant animals eliminates the accumulation of protein inclusions in salivary gland cells.
Expression of BacA\p35Scer\UAS.cHa under the control of Scer\GAL4da.G32 in ctpG0371 mutant animals results in almost complete inhibition of salivary gland degradation, and 96% of animals display intact salivary glands.
Larval motility defects observed in animals expressing Hsap\ARQ52.Scer\UAS in motor neurons under the control of Scer\GAL4D42 and fed on food supplemented with DHT, is enhanced in a ctpG0371 mutant background.
ctpG0371 is rescued by Scer\GAL4fkh.PH/ctpUAS.cBa
ctpG0371 is partially rescued by ctpUAS.cBa/Scer\GAL4da.G32
Expression of ctpScer\UAS.cBa under the control of Scer\GAL4fkh.PH in ctpG0371 mutant animals rescues the defects in salivary gland degradation. Most animals have no salivary glands, with a small number showing fragmented salivary glands.
Expression of ctpScer\UAS.cBa throughout development under the control of Scer\GAL4da.G32 rescues the viability of 63% of ctpG0371 adult males.
P{lacW} not verified as causing the lethality.