Polytene chromosomes normal.
Amino acid replacement: R?C. The amino acid replacement is at a phosphate contacting Arginine residue within the second zinc finger of the DNA binding domain.
Missense mutation in the DNA-binding domain.
Nucleotide substitution: C?T. Amino acid replacement: R130C. Mutation is in the conserved arginine residues in the DNA binding domain (DBD).
C2040526T
R130C | usp-PA; R130C | usp-PB
Homozygous clones in the eye imaginal disc that lie posterior to the morphogenetic furrow have an abnormal arrangement of developing photoreceptor clusters. The clusters are irregularly spaced and differentiate prematurely, although they contain the normal number of cells. Movement of the morphogenetic furrow is accelerated where it passes through a homozygous clone. This displacement of the furrow is not caused by excess proliferation of usp4 cells in the clone.
Heterozygotes display thoracic defects ranging from mild, slight separation of the microchaetes and macrochaetes along the dorsal midline, to severe, a cleft extending through both the notum and scutellum. Flies also have bent and misshapen sensory bristles and develop severely knarled legs.
Females with usp mutant germ cells give rise to fully viable and fertile female usp mutant heterozygotes, but non-viable usp mutant/Y males. Most of these males die just prior to hatching, with the rest dying shortly after. The unhatched embryos have cuticular scarring in the region posterior of A9. In contrast usp mutant/Y males derived from germ cells with functional usp hatch and die between the first and second instar periods, with no gross cuticular defects. usp4 mosaic females mutant in the germline lay very few unfertilised eggs, in contrast to usp4 homozygous females (mutant both soma and germline), indicating that the fertilisation defect in these homozygotes is due to lack of usp in the soma.
Lethality occurs during embryonic and larval stages. Phenotype of homozygous germ line clones is maternal effect lethal.
Homozygous usp4 germline generates misshapen eggs.
usp4 has lethal | larval stage phenotype, suppressible by usp::Cten\Usp-1d-c
Df(2L)let-7-CKO1, P{W8,let7CΔmir-let7}, usp4 has abnormal neuroanatomy phenotype
EcR[+]/EcRQ50st, usp4 has abnormal neuroanatomy phenotype
Scer\GAL4αTub84B.PL, ari-1UAS.cFa, usp4/usp[+] has lethal | embryonic stage phenotype
Scer\GAL4αTub84B.PL, ari-13747, ari-1UAS.cFa, usp4/usp[+] has lethal | second instar larval stage phenotype
Df(2L)let-7-CKO1, P{W8,let7CΔmir-let7}, usp4 has lobe system of adult mushroom body phenotype
EcR[+]/EcRQ50st, usp4 has lobe system of adult mushroom body phenotype
EcR[+]/EcRQ50st, usp4 has adult mushroom body beta-lobe phenotype
usp4/+ ; Df(2L)let-7-CKO1/+ ; P{W8,let-7-CΔlet-7} animals have defects in the mushroom body α/β lobes: the lobes may appear slim.
usp4/+ ; EcRQ50st/+ animals have defects in the mushroom body α/β lobes: the lobes may appear slim or the β lobes may be fused.
Mutant larvae rescued by one copy of usp::Cten\Usp-1d-c die at the late third instar stage. Rescued usp4 mutants show a cessation of movement during the late third instar, but larvae show no sign of prepupal tanning except along the denticle belts of the midsegments. The addition of a second copy of usp::Cten\Usp-1d-c leads to further tanning of larvae, but they continue to fail to undergo pupal contraction, and anterior spiracle eversion is incomplete. The addition of three and four copies does allow some adults to survive.