Compared to sibling heterozygotes, BHDnull/BHDnull larvae are smaller (including reduced size of imaginal discs and brains), survive for a prolonged period of time as larvae, have reduced numbers of mitotic cells, have reduced DNA synthesis, have thin fat bodies with cells that contain vacuoles and shrunken nuclei, and have reduced cell volumes and DNA contents of salivary glands. Compared to sibling heterozygotes, BHDnull/BHDnull larvae become even smaller when on less nutritious (yeast-free) food.
Even under normal feeding conditions, autophagy in the fat bodies is elevated in BHDnull/BHDnull larvae compared to heterozygotes; suppressing autophagy by feeding with an autophagy inhibitor does not restore normal growth or viability.
Growth delay of BHDnull/BHDnull larvae is significantly rescued by yeast-rich or leucine-added food, though mutants still die at the pupal stage; addition of rapamycin (inhibitor of mTOR signaling) suppresses these rescue effects.
BHDnull/BHDnull mutant clones in imaginal discs or eye discs are similar in size and do not show clear cell number differences to wild type, suggesting BHD is not required cell-autonomously.
BHDnull has lethal - all die before end of larval stage | recessive phenotype, suppressible | partially by Hsap\FLCNUAS.cLa/Scer\GAL4hs.PU
Hsap\FLCNScer\UAS.cLa driven by Scer\GAL4hs.PU partially rescues BHDnull/BHDnull larval lethality (about one third develop into pupae).
BHDC.T:Avic\GFP-EGFP or BHDN.T:Avic\GFP-EGFP rescues BHDnull/BHDnull larval lethality.