2bp deletion within the first exon of Svip; this results in a frame-shift and a premature stop codon upstream of the VCP-interaction motif.
A 2bp deletion in the first exon that results in a frameshift and premature stop codon.
SvipKO homozygosity leads to fragmentation of the lysosome tubule network in body wall muscles of third instar larvae, as well as in adult abdominal muscles: decreased number of junctions per tubule and decreased tubule density.
SvipKO homozygous adults show a progressive decrease in climbing activity; aged adults show shorter neuromuscular junctions, which have a darkened appearance with large vesicles; abdominal muscles show signs of wasting, impaired mitochondria morphology and hallmarks of neuromuscular synapse degeneration (less striated t-tubules and disorganized Z-bands, nuclei are variably positioned, mitochondria are altered and with apparent vacuoles).
TTM motoneurons show progressive degeneration from pupal development through adult life, with a progressive decrease in dendrite length and volume; TTM and DLM motoneuron neuromuscular junction recordings show failure in aged adults. Aged adults show increased apoptosis in the ventral nerve cord, optic lobe and neural circuitry of the head.
SvipKO has lysosome phenotype, suppressible | partially by spinUAS.Tag:FKBP(F36V),mCherry/TER94sfGFP
SvipKO has somatic muscle cell phenotype, suppressible | partially by spinUAS.Tag:FKBP(F36V),mCherry/TER94sfGFP
SvipKO heterozygous third instar larvae expressing SvipS82L.UAS under the control of Scer\GAL4Mhc.PU show fragmentation of the lysosome tubule network: decreased number of junctions per tubule and decreased tubule density.
SvipKO is partially rescued by SvipUAS.cJa/Scer\GAL4Mhc.PU
SvipKO is not rescued by SvipRR.UAS.mCherry/Scer\GAL4Mhc.PU
SvipKO is not rescued by Scer\GAL4unspecified/SvipUAS.cJa