Mutation in the translation start codon and a frame shift in the HDAC6 open reading frame.
AGATCT
Mutation in ATG codon and frameshift introduced by homologous recombination. AAC[ATG]GT (start codon indicated) replaced with AGATCT.
HDAC6KO mutant larvae show a significantly increased response to gentle touch, as compared to controls.
HDAC6KO mutant third instar larvae display mild but significant increase in the density of perinuclear microtubule network in muscle cells compared to controls at room temperature and their microtubules (MT) are also more resistant to cold-induced destabilization (the perinuclear MTs are significantly denser in HDAC6KO mutant muscle cells than in wild-type after cold treatment).
HDAC6KO mutants do not exhibit macro-morphological changes in synaptic features, with the number of T-bars, synaptic vesicles, or mitochondria per boutonic area, not differing from controls. However, using serial-section election microscopy, HDAC6KO mutant T-bars exhibit significantly smaller 'top sizes' and fewer tethered synaptic vesicles compared to controls.
At low-frequency stimulation, the excitatory junctional current amplitude in HDAC6KO mutants is marginally smaller.
Homozygotes show no obvious morphological abnormalities under normal culture conditions.
Mutants do not show any notable difference to wild-type animals in response to oxidative or heat stress.
Mutant adults do not show defects in climbing ability (assayed up to 20 days after eclosion).
HDAC6KO has abnormal touch response | larval stage phenotype, non-enhanceable by αTub84BK40A/αTub84BK40A
HDAC6KO has abnormal touch perception phenotype, non-enhanceable by αTub84BK40A/αTub84BK40A
HDAC6KO has short lived | nutrition conditional phenotype, suppressible | RU486 conditional by Scer\GAL4Mhc.Switch.PO/Lsd-2HMS00629
HDAC6KO has abnormal locomotor behavior | adult stage | progressive phenotype, suppressible by Lsd-2KG00149
HDAC6KO has short lived | nutrition conditional phenotype, suppressible by Lsd-2KG00149
HDAC6KO is an enhancer of male semi-sterile phenotype of Sirt25B-2-35
HDAC6KO is an enhancer of decreased fecundity | male phenotype of Sirt25B-2-35
HDAC6KO is an enhancer of abnormal neuroanatomy | adult stage | conditional phenotype of Hsap\SNCAUAS.cFa, Scer\GAL4ple.PF
HDAC6KO is an enhancer of abnormal locomotor behavior | adult stage | conditional phenotype of Hsap\SNCAUAS.cFa, Scer\GAL4ple.PF
HDAC6KO is an enhancer of abnormal neuroanatomy | adult stage | conditional phenotype of Hsap\SNCAUAS.cFa, Scer\GAL4GMR.PU
HDAC6KO is a non-enhancer of male semi-sterile phenotype of Cep97Δ
HDAC6KO is a non-enhancer of abnormal touch response | third instar larval stage phenotype of Cep97Δ
HDAC6KO is a non-enhancer of abnormal locomotor behavior phenotype of Cep97Δ
HDAC6KO is a non-enhancer of decreased fecundity | male phenotype of Cep97Δ
HDAC6KO is a non-enhancer of abnormal touch response | third instar larval stage phenotype of Sirt25B-2-35
HDAC6KO is a suppressor of abnormal locomotor behavior phenotype of Sirt25B-2-35
HDAC6KO/HDAC6[+] is a suppressor of abnormal locomotor behavior | adult stage phenotype of Hsap\TARDBPA315T.UAS.cVa, Scer\GAL4elav.PU
HDAC6KO is a non-suppressor of abnormal touch response | third instar larval stage phenotype of Sirt25B-2-35
HDAC6KO is a non-suppressor of male semi-sterile phenotype of Cep97Δ
HDAC6KO is a non-suppressor of abnormal touch response | third instar larval stage phenotype of Cep97Δ
HDAC6KO is a non-suppressor of abnormal locomotor behavior phenotype of Cep97Δ
HDAC6KO is a non-suppressor of decreased fecundity | male phenotype of Cep97Δ
HDAC6KO, Sirt25B-2-35 has viable phenotype
HDAC6KO has wing disc | third instar larval stage phenotype, non-enhanceable by Sirt25B-2-35
HDAC6KO has centriole phenotype, non-enhanceable by Sirt25B-2-35
HDAC6KO has spermatozoon phenotype, non-enhanceable by Cep97Δ
HDAC6KO has spermatid axoneme | P-stage phenotype, non-enhanceable by Cep97Δ
HDAC6KO has mesothoracic tergotrochanter muscle cell | progressive phenotype, suppressible by Scer\GAL4Mhc.PK/Hsc70-4UAS.cEa
HDAC6KO has lipid droplet | adult stage | progressive phenotype, suppressible by Scer\GAL4Mhc.PK/Hsc70-4UAS.cEa
HDAC6KO has mesothoracic tergotrochanter muscle cell | progressive phenotype, suppressible by Scer\GAL4Mhc.PK/Lsd-2HMS00629
HDAC6KO has lipid droplet | adult stage | progressive phenotype, suppressible by Scer\GAL4Mhc.PK/Lsd-2HMS00629
HDAC6KO has mesothoracic tergotrochanter muscle cell | progressive phenotype, suppressible by Lsd-2HMS00629
HDAC6KO has lipid droplet | adult stage | progressive phenotype, suppressible by Lsd-2KG00149
HDAC6KO has spermatozoon phenotype, non-suppressible by Cep97Δ
HDAC6KO has spermatid axoneme | P-stage phenotype, non-suppressible by Cep97Δ
HDAC6KO has wing disc | third instar larval stage phenotype, non-suppressible by Sirt25B-2-35
HDAC6KO has centriole phenotype, non-suppressible by Sirt25B-2-35
HDAC6KO has mesothoracic tergotrochanter muscle cell | progressive phenotype, non-suppressible by Hsc70-4K71S.UAS/Scer\GAL4Mhc.PK
HDAC6KO has lipid droplet | adult stage | progressive phenotype, non-suppressible by Hsc70-4K71S.UAS/Scer\GAL4Mhc.PK
HDAC6KO has mesothoracic tergotrochanter muscle cell | progressive phenotype, non-suppressible by Scer\GAL4Mhc.PK/Hsc70-4D206S.UAS
HDAC6KO has lipid droplet | adult stage | progressive phenotype, non-suppressible by Scer\GAL4Mhc.PK/Hsc70-4D206S.UAS
HDAC6KO has mesothoracic tergotrochanter muscle cell | progressive phenotype, non-suppressible by Scer\GAL4Mhc.PK/DnaJ-1UAS.cKa
HDAC6KO has lipid droplet | adult stage | progressive phenotype, non-suppressible by Scer\GAL4Mhc.PK/DnaJ-1UAS.cKa
HDAC6KO has mesothoracic tergotrochanter muscle cell | progressive phenotype, non-suppressible by mTorGL00156/Scer\GAL4Mhc.PK
HDAC6KO has lipid droplet | adult stage | progressive phenotype, non-suppressible by mTorGL00156/Scer\GAL4Mhc.PK
HDAC6KO has mesothoracic tergotrochanter muscle cell | progressive phenotype, non-suppressible by Scer\GAL4Mhc.PK/Atg8aUASp.EGFP,mCherry
HDAC6KO has lipid droplet | adult stage | progressive phenotype, non-suppressible by Scer\GAL4Mhc.PK/Atg8aUASp.EGFP,mCherry
HDAC6KO has mesothoracic tergotrochanter muscle cell | progressive phenotype, non-suppressible by Scer\GAL4Mhc.PK/HslUAS.EGFP
HDAC6KO has lipid droplet | adult stage | progressive phenotype, non-suppressible by Scer\GAL4Mhc.PK/HslUAS.EGFP
HDAC6KO has mesothoracic tergotrochanter muscle cell | progressive phenotype, non-suppressible by Scer\GAL4Mhc.PK/bmmUAS.cUa
HDAC6KO has lipid droplet | adult stage | progressive phenotype, non-suppressible by Scer\GAL4Mhc.PK/bmmUAS.cUa
HDAC6KO has mesothoracic tergotrochanter muscle cell | progressive phenotype, non-suppressible by Scer\GAL4Mhc.PK/LpinHMJ30181
HDAC6KO has lipid droplet | adult stage | progressive phenotype, non-suppressible by Scer\GAL4Mhc.PK/LpinHMJ30181
HDAC6KO has mesothoracic tergotrochanter muscle cell | progressive phenotype, non-suppressible by Lsd-138
HDAC6KO has lipid droplet | adult stage | progressive phenotype, non-suppressible by Lsd-138
HDAC6KO has mesothoracic tergotrochanter muscle cell | progressive phenotype, non-suppressible by Scer\GAL4NP5440/Lsd-2HMS00629
HDAC6KO has lipid droplet | adult stage | progressive phenotype, non-suppressible by Scer\GAL4NP5440/Lsd-2HMS00629
HDAC6KO is an enhancer of dopaminergic neuron | conditional phenotype of Hsap\SNCAUAS.cFa, Scer\GAL4ple.PF
HDAC6KO is an enhancer of ommatidium | conditional phenotype of Hsap\SNCAUAS.cFa, Scer\GAL4GMR.PU
HDAC6KO is an enhancer of eye photoreceptor cell | conditional phenotype of Hsap\SNCAUAS.cFa, Scer\GAL4GMR.PU
HDAC6KO is a non-enhancer of spermatozoon phenotype of Cep97Δ
HDAC6KO is a non-enhancer of spermatid axoneme | P-stage phenotype of Cep97Δ
HDAC6KO is a non-enhancer of wing disc | third instar larval stage phenotype of Cep97Δ
HDAC6KO is a non-enhancer of wing disc | third instar larval stage phenotype of Sirt25B-2-35
HDAC6KO is a non-enhancer of centriole phenotype of Sirt25B-2-35
HDAC6KO is a suppressor of primary spermatocyte | P-stage phenotype of Cep97Δ
HDAC6KO/HDAC6[+] is a suppressor of t-bar phenotype of Hsap\TARDBPA315T.UAS.cVa, Scer\GAL4elav.PU
HDAC6KO is a suppressor of microtubule phenotype of Hsap\MAPTUAS.cWa, Scer\GAL4C57
Df(1)ED7294/HDAC6KO is a suppressor of microtubule phenotype of Hsap\MAPTUAS.cWa, Scer\GAL4C57
HDAC6KO is a suppressor of microtubule phenotype of Hsap\MAPTV337M.UAS, Scer\GAL4C57
Df(1)ED7294/HDAC6KO is a suppressor of microtubule phenotype of Hsap\MAPTV337M.UAS, Scer\GAL4C57
HDAC6KO is a suppressor of embryonic/larval neuromuscular junction | larval stage phenotype of Hsap\MAPTV337M.UAS, Scer\GAL4elav.PU
HDAC6KO is a suppressor of NMJ bouton | larval stage phenotype of Hsap\MAPTV337M.UAS, Scer\GAL4elav.PU
HDAC6KO is a suppressor of embryonic/larval neuromuscular junction | larval stage phenotype of Hsap\MAPTUAS.cWa, Scer\GAL4elav.PU
HDAC6KO is a suppressor of NMJ bouton | larval stage phenotype of Hsap\MAPTUAS.cWa, Scer\GAL4elav.PU
HDAC6KO is a non-suppressor of spermatid axoneme | P-stage phenotype of Cep97Δ
HDAC6KO is a non-suppressor of wing disc | third instar larval stage phenotype of Cep97Δ
HDAC6KO is a non-suppressor of wing disc | third instar larval stage phenotype of Sirt25B-2-35
HDAC6KO is a non-suppressor of centriole phenotype of Sirt25B-2-35
HDAC6KO is a non-suppressor of spermatozoon phenotype of Cep97Δ
HDAC6KO is a non-suppressor of microtubule phenotype of Scer\GAL4C57, spasUAS.cTa
The decrease in perinuclear microtubule density which is seen in larval muscle cells expressing spasScer\UAS.cTa under the control of Scer\GAL4C57 is not altered by the presence of HDAC6KO.
The decrease in perinuclear microtubule density which is seen in larval muscle cells expressing Kat60Scer\UAS.cMa under the control of Scer\GAL4C57 is not altered by the presence of HDAC6KO. The microtubule fibres are significantly shorter in the double mutant cells.
A HDAC6KO heterozygous background suppresses the larger synaptic T-bar size found upon expression of Hsap\TARDBPA315T.cMa.Scer\UAS under the control of Scer\GAL4elav.PU.
A HDAC6KO heterozygous background suppresses the larger synaptic T-bar size found upon expression of Hsap\TARDBPA382T.cMa.Scer\UAS under the control of Scer\GAL4elav.PU.
While heterozygosity for HDAC6KO alone has no effect on activity or negative geotaxis, these conditions both significant suppress the activity and geotaxis defects in flies expressing Hsap\TARDBPA315T.cMa.Scer\UAS under the control of Scer\GAL4elav.PU.
HDAC6KO and HDAC6KO/Df(1)ED7294 each rescue the defects in perinuclear microtubule density seen in the muscles of animals expressing either Hsap\MAPTScer\UAS.cWa or Hsap\MAPTV337M.Scer\UAS under the control of Scer\GAL4C57.
HDAC6KO completely suppresses the increase in the number of satellite boutons at the neuromuscular junction (NMJ) and the decrease in average NMJ length caused by expression of either Hsap\MAPTScer\UAS.cWa or Hsap\MAPTV337M.Scer\UAS under the control of Scer\GAL4elav.PU.
HDAC6KO enhances the loss of dopaminergic (DA) neurons caused by expression of Hsap\SNCAScer\UAS.cFa under the control of Scer\GAL4ple.PF; the neuron degeneration phenotype is apparent at 10 days after eclosion in the double mutant flies, and 20 days after eclosion it is more severe than that seen in the Hsap\SNCAScer\UAS.cFa, Scer\GAL4ple.PF single mutants.
HDAC6KO enhances the retinal degeneration phenotype seen in adults expressing Hsap\SNCAScer\UAS.cFa under the control of Scer\GAL4GMR.PU.
HDAC6KO enhances the climbing ability defect caused by expression of Hsap\SNCAScer\UAS.cFa under the control of Scer\GAL4ple.PF; defects are seen 10 days after eclosion in the double mutant flies (defects are not seen until 20 days after eclosion in Hsap\SNCAScer\UAS.cFa, Scer\GAL4ple.PF single mutants) and the defects at 20 days after eclosion are more severe in the double mutants.
HDAC6KO decreases the number of Lewy body-like Hsap\SNCA positive inclusions seen in the brains of adults expressing Hsap\SNCAScer\UAS.cFa under the control of Scer\GAL4ple.PF such that no inclusions are seen in 10 day old flies and the number of inclusions seen in 20 days old flies is reduced.
HDAC6KO is rescued by Scer\GAL4Mhc.PK/HDAC6UAS.cDa
HDAC6KO is rescued by HDAC6UAS.cDa/Scer\GAL4C57
HDAC6KO is rescued by Scer\GAL4C57/HDAC6H237A.UAS
HDAC6KO is partially rescued by Scer\GAL4Mhc.PK/HDAC6N.UAS
HDAC6KO is partially rescued by HDAC6H664A.UAS/Scer\GAL4C57
HDAC6KO is not rescued by HDAC6UAS.cDa/Scer\GAL4NP5440
HDAC6KO is not rescued by Scer\GAL4Mhc.PK/HDAC6H237A.H664A.UAS
HDAC6KO is not rescued by HDAC6H237A.H664A.UAS/Scer\GAL4C57
Expression of either HDAC6Scer\UAS.cDa or HDAC6H237A.Scer\UAS under the control of Scer\GAL4C57 completely suppresses the ability of HDAC6KO to rescue the perinuclear microtubule density defects seen in the muscles of animals expressing either Hsap\MAPTV337M.Scer\UAS under the control of Scer\GAL4C57.
Expression of HDAC6H664A.Scer\UAS under the control of Scer\GAL4C57 partially suppresses the ability of HDAC6KO to rescue the perinuclear microtubule density defects seen in the muscles of animals expressing either Hsap\MAPTV337M.Scer\UAS under the control of Scer\GAL4C57.
Expression of HDAC6H237A.H664A.Scer\UAS under the control of Scer\GAL4C57 does not affect the ability of HDAC6KO to rescue the perinuclear microtubule density defects seen in the muscles of animals expressing either Hsap\MAPTV337M.Scer\UAS under the control of Scer\GAL4C57.