FlyBase Allele Report: Hsap\HTT[Q138.UAS.mRFP1]

General Information

Symbol

Hsap\HTT^{Q138.UAS.mRFP1}

Species

H. sapiens

Name

FlyBase ID

FBal0267405

Feature type

allele

Associated gene

Hsap\HTT

Associated Insertion(s)

Carried in Construct

P{UAS-HTT.Q138.mRFP}

Also Known As

UAS-HttQ138-mRFP, UAS-htt.138Q.mRFP, UAS-RFP-HttQ138

Key Links

Transgenic product class

inframe_deletion

(Weiss et al., 2012)

trinucleotide_repeat_expansion

(Weiss et al., 2012)

Mutagen

in vitro construct

Nature of the Allele

Transgenic product class

inframe_deletion

(Weiss et al., 2012)

trinucleotide_repeat_expansion

(Weiss et al., 2012)

Mutagen

in vitro construct

(Weiss et al., 2012)

Progenitor genotype

Carried in construct

P{UAS-HTT.Q138.mRFP}

Cytology

Description

UASt sequences drive expression of the N terminal 588 amino acids of Hsap\HTT cDNA containing a pathogenic tract of 138 polyQ repeats. The construct is tagged at the N-terminal end with mRFP1.

(Weiss et al., 2012)

Allele components

Component

Use(s)

Regulatory region(s)

UASt

binary expression system - regulatory region

Encoded product / tool

Hsap\HTT

Tagged with

mRFP1

red fluorescent protein

Mutations Mapped to the Genome

Curation Data

Type

Location

Additional Notes

References

Variant Molecular Consequences

Associated Sequence Data

DDBJ / EMBL / GenBank

DNA sequence

Protein sequence

UniProtKB/Swiss-Prot

UniProtKB/TrEMBL

Expression Data

Reporter Expression

Additional Information

Statement

Reference

Marker for

Reflects expression of

Reporter construct used in assay

Human Disease Associations

Disease Ontology (DO) Annotations

Models Based on Experimental Evidence ( 1 )

Disease

Evidence

References

model of Huntington's disease

CEA

(Swinter et al., 2023, Huelsmeier et al., 2021, Tandon and Sarkar, 2021, Joag et al., 2020, Hansen et al., 2019, Raj and Sarkar, 2019, Zhu et al., 2019, Akbergenova and Littleton, 2017, Krench et al., 2016, Weiss and Littleton, 2016, Babcock and Ganetzky, 2015, Weiss et al., 2012)

Modifiers Based on Experimental Evidence ( 1 )

Disease

Interaction

References

model of Huntington's disease

is ameliorated by orb2^UAS.A.EGFP

(Joag et al., 2020)

is ameliorated by orb2^UAS.B.EGFP

(Joag et al., 2020)

is exacerbated by orb2^RNAi.UAS.cUa

(Joag et al., 2020)

is ameliorated by Rab7^T22N.UAS

(Akbergenova and Littleton, 2017)

is ameliorated by Snx16^Δ1

(Akbergenova and Littleton, 2017)

is ameliorated by Snx16^Δ2

(Akbergenova and Littleton, 2017)

is ameliorated by gbb¹

(Akbergenova and Littleton, 2017)

is ameliorated by gbb²

(Akbergenova and Littleton, 2017)

is ameliorated by InR^UAS.Exel

(Tandon and Sarkar, 2021, Raj and Sarkar, 2019)

is ameliorated by Pi3K92E^CAAX.UAS

(Hansen et al., 2019)

is exacerbated by Pi3K92E^{D954A.UAS.Tag:MYC}

(Hansen et al., 2019)

is exacerbated by Nmnat^unspecified

(Zhu et al., 2019)

is ameliorated by Nmnat^UAS.cZa

(Zhu et al., 2019)

is ameliorated by Atx2^{KI.ΔcIDR-GFP}

(Huelsmeier et al., 2021)

is ameliorated by Hsap\HSPA1L^UAS.cWa

(Swinter et al., 2023)

is ameliorated by Lkb1^4A4-2

(Schulte et al., 2011)

Comments on Models/Modifiers Based on Experimental Evidence ( 0 )

Disease-implicated variant(s)

This allele represents a human variant implicated in disease.

(Weiss et al., 2012)

HTT:p.Gln18[138]

(FlyBase curators, 2019-)

Variants Synonym(s)

HTT:p.Gln18[138Gln]

HTT, (CAG)n REPEAT EXPANSION

Associated human disease model(s)

Huntington disease

External database links

ClinVar: HTT_409

Comments concerning this variant

Phenotypic Data

Phenotypic Class

abnormal grooming behavior | adult stage, with Scer\GAL4^elav.PLu

(Weiss et al., 2012)

abnormal locomotor behavior | adult stage, with Scer\GAL4^elav.PLu

(Weiss et al., 2012)

abnormal neuroanatomy | adult stage | cell non-autonomous | progressive, with Scer\GAL4^Orco.PK

(Babcock and Ganetzky, 2015)

abnormal neuroanatomy | adult stage | progressive, with Scer\GAL4^Orco.PK

(Babcock and Ganetzky, 2015)

increased cell death | adult stage | cell non-autonomous | progressive, with Scer\GAL4^Orco.PK

(Babcock and Ganetzky, 2015)

increased cell death | adult stage | progressive, with Scer\GAL4^Orco.PK

(Babcock and Ganetzky, 2015)

increased cell death | larval stage, with Scer\GAL4^Appl.G1a

(Swinter et al., 2023)

lethal - all die before end of P-stage, with Scer\GAL4^elav-C155

(Weiss et al., 2012)

lethal - all die before end of pupal stage, with Scer\GAL4^elav-C155

(Schulte et al., 2011)

long lived | chemical conditional, with Scer\GAL4^elav-C155

(Schulte et al., 2011)

partially lethal - majority die, with Scer\GAL4^elav-C155

(Weiss et al., 2012)

some die during pharate adult stage, with Scer\GAL4^elav-C155

(Weiss et al., 2012)

some die during pupal stage, with Scer\GAL4^elav-C155

(Schulte et al., 2011)

viable, with Scer\GAL4^elav.PLu

(Weiss et al., 2012)

Phenotype Manifest In

adult cerebrum | cell non-autonomous | progressive, with Scer\GAL4^Orco.PK

(Babcock and Ganetzky, 2015)

adult optic lobe | cell non-autonomous | progressive, with Scer\GAL4^Orco.PK

(Babcock and Ganetzky, 2015)

axon, with Scer\GAL4^elav-C155

(Schulte et al., 2011)

axon | larval stage, with Scer\GAL4^Appl.G1a

(Swinter et al., 2023)

larval segmental nerve, with Scer\GAL4^Appl.G1a

(Swinter et al., 2023)

mitochondrion | larval stage, with Scer\GAL4^Appl.G1a

(Swinter et al., 2023)

neuromere, with Scer\GAL4^elav-C155

(Schulte et al., 2011)

neuron, with Scer\GAL4^elav-C155

(Schulte et al., 2011)

olfactory receptor neuron | adult stage, with Scer\GAL4^Orco.PK

(Babcock and Ganetzky, 2015)

Detailed Description

Statement

Reference

Expression of Hsap\HTT^{Q138.Scer\UAS.T:Disc\RFP-mRFP} driven by Scer\GAL4^Orco.PK only in adulthood (in combination with TubGAL80ts) leads to protein aggregates in olfactory receptor neuron terminals in 1 day old flies (but no evidence of cell death); after 15 days, cell death is evident in the central brain and optic lobes and after 30 days cell death is even more widespread.

(Babcock and Ganetzky, 2015)

Expression of Hsap\HTT^{Q138.Scer\UAS.T:Disc\RFP-mRFP} under the control of Scer\GAL4^elav-C155 causes pharate adult lethality, with less than 1% viable adult escapers. Expression of Hsap\HTT^{Q138.Scer\UAS.T:Disc\RFP-mRFP} with a weaker elav-GAL4 driver (Scer\GAL4^elav.PLu) results in viable adults that appear behaviourally normal at the time of eclosion. However, several days after eclosion these adults begin to exhibit motor coordination defects and abnormal grooming behaviours, worsening with age and resulting in premature death. The time taken for 50% of flies to die in the P{UAS-HTT.Q138.mRFP}A insertion line is 70% shorter than in controls, although this is reduced to 30% in the lower expressing P{UAS-HTT.Q138.mRFP}B insertion line.

Third instar larvae expressing Hsap\HTT^{Q138.Scer\UAS.T:Disc\RFP-mRFP} under the control of Scer\GAL4^elav-C155 form distinct cytoplasmic aggregates throughout the cytoplasm and neurites of neurons in both the CNS and PNS. Expressing Hsap\HTT^{Q138.Scer\UAS.T:Disc\RFP-mRFP} ubiquitously under the control of Scer\GAL4^αTub84B.PL shows that aggregates also form in non-neuronal cells, including epidermis and salivary glands. No nuclear aggregates were observed in any cell type. FRAP microscopy of live anaesthetised third instar larval axons expressing Hsap\HTT^{Q138.Scer\UAS.T:Disc\RFP-mRFP} under the control of Scer\GAL4^Ccap.PP shows that the aggregates continue to accumulate protein following photobleaching, with larger aggregates recovering more quickly than smaller ones. Restricting Hsap\HTT^{Q138.Scer\UAS.T:Disc\RFP-mRFP} expression until the larvae reach the third instar larval stage shows that existing neuronal aggregates increase in size up to 12 hours, after which new aggregates begin to form. Restricting expression of Hsap\HTT^{Q138.Scer\UAS.T:Disc\RFP-mRFP} after aggregates have formed leads to a reduction in aggregate size and number.

(Weiss et al., 2012)

Hsap\HTT^{Q138.Scer\UAS.T:Disc\RFP-mRFP} expressing neurons display morphological indicators of reduced neuronal health, including smaller neuromeres, increased branching, and reduced axonal connectivity.

Addition of rapamycin, 18β-Glycyrrhetinic acid, Carbenoxolone, Camptothecin or 10-Hydroxycamptothecin increases the longevity of larvae pan-neuronally expressing Hsap\HTT^{Q138.Scer\UAS.T:Disc\RFP-mRFP} under the control of Scer\GAL4^elav-C155.

(Schulte et al., 2011)

External Data

Linkouts

Interactions

Show genetic interaction network for Enhancers & Suppressors

Phenotypic Class

Suppressed by

Statement

Reference

Hsap\HTT^{Q138.UAS.mRFP1}, Scer\GAL4^elav-C155 has lethal | pharate adult stage phenotype, suppressible | partially by +/Df(3L)brm11

(Weiss et al., 2012)

Hsap\HTT^{Q138.UAS.mRFP1}, Scer\GAL4^elav-C155 has lethal | pharate adult stage phenotype, suppressible | partially by Df(3R)H-B79/+

(Weiss et al., 2012)

Hsap\HTT^{Q138.UAS.mRFP1}, Scer\GAL4^elav-C155 has lethal | pharate adult stage phenotype, suppressible | partially by Df(3L)BSC33/+

(Weiss et al., 2012)

Hsap\HTT^{Q138.UAS.mRFP1}, Scer\GAL4^elav-C155 has lethal | pharate adult stage phenotype, suppressible | partially by Df(2R)59AB/+

(Weiss et al., 2012)

Hsap\HTT^{Q138.UAS.mRFP1}, Scer\GAL4^elav-C155 has lethal | pharate adult stage phenotype, suppressible | partially by Df(2R)sple-J1/+

(Weiss et al., 2012)

Hsap\HTT^{Q138.UAS.mRFP1}, Scer\GAL4^elav-C155 has lethal | pharate adult stage phenotype, suppressible | partially by Df(3L)ED220/+

(Weiss et al., 2012)

Hsap\HTT^{Q138.UAS.mRFP1}, Scer\GAL4^elav-C155 has lethal | pharate adult stage phenotype, suppressible | partially by Df(3R)MAP117/+

(Weiss et al., 2012)

Hsap\HTT^{Q138.UAS.mRFP1}, Scer\GAL4^elav-C155 has lethal | pharate adult stage phenotype, suppressible | partially by Df(3R)MAP2/+

(Weiss et al., 2012)

Hsap\HTT^{Q138.UAS.mRFP1}, Scer\GAL4^elav-C155 has lethal | pharate adult stage phenotype, suppressible | partially by hzg^c02324/CG5830[+]

(Weiss et al., 2012)

Hsap\HTT^{Q138.UAS.mRFP1}, Scer\GAL4^elav-C155 has lethal | pharate adult stage phenotype, suppressible | partially by lab[+]/lab¹⁴

(Weiss et al., 2012)

Hsap\HTT^{Q138.UAS.mRFP1}, Scer\GAL4^elav-C155 has lethal | pharate adult stage phenotype, suppressible by Df(3L)vin7/+

(Weiss et al., 2012)

Hsap\HTT^{Q138.UAS.mRFP1}, Scer\GAL4^elav-C155 has lethal | pharate adult stage phenotype, suppressible | partially by Scr[+]/Scr^CP1

(Weiss et al., 2012)

Hsap\HTT^{Q138.UAS.mRFP1}, Scer\GAL4^elav-C155 has lethal | pharate adult stage phenotype, suppressible | partially by Scr⁶/Scr[+]

(Weiss et al., 2012)

Hsap\HTT^{Q138.UAS.mRFP1}, Scer\GAL4^elav-C155 has lethal | pharate adult stage phenotype, suppressible | partially by pb¹/pb[+]

(Weiss et al., 2012)

Hsap\HTT^{Q138.UAS.mRFP1}, Scer\GAL4^elav-C155 has lethal | pharate adult stage phenotype, suppressible | partially by Dfd[+]/Dfd⁶

(Weiss et al., 2012)

Hsap\HTT^{Q138.UAS.mRFP1}, Scer\GAL4^elav-C155 has lethal | pharate adult stage phenotype, suppressible | partially by Ubx[+]/Ubx⁵¹

(Weiss et al., 2012)

Hsap\HTT^{Q138.UAS.mRFP1}, Scer\GAL4^elav-C155 has lethal | pharate adult stage phenotype, suppressible | partially by Df(2L)JS17/+

(Weiss et al., 2012)

Hsap\HTT^{Q138.UAS.mRFP1}, Scer\GAL4^elav-C155 has lethal | pharate adult stage phenotype, suppressible | partially by Df(2R)cn9/+

(Weiss et al., 2012)

Hsap\HTT^{Q138.UAS.mRFP1}, Scer\GAL4^elav-C155 has lethal | pharate adult stage phenotype, suppressible | partially by Df(2R)AA21/+

(Weiss et al., 2012)

Hsap\HTT^{Q138.UAS.mRFP1}, Scer\GAL4^elav-C155 has lethal | pharate adult stage phenotype, suppressible | partially by +/Df(2R)59AD

(Weiss et al., 2012)

Hsap\HTT^{Q138.UAS.mRFP1}, Scer\GAL4^elav-C155 has lethal | pharate adult stage phenotype, suppressible | partially by Df(2L)BSC109/+

(Weiss et al., 2012)

Hsap\HTT^{Q138.UAS.mRFP1}, Scer\GAL4^elav-C155 has lethal | pharate adult stage phenotype, suppressible | partially by Df(3R)Tpl10/+

(Weiss et al., 2012)

Hsap\HTT^{Q138.UAS.mRFP1}, Scer\GAL4^elav-C155 has lethal | pharate adult stage phenotype, suppressible | partially by Df(3L)st-f13/+

(Weiss et al., 2012)

Hsap\HTT^{Q138.UAS.mRFP1}, Scer\GAL4^elav-C155 has lethal | pupal stage phenotype, suppressible by Lkb1^4B1-11/lkb1[+]

(Schulte et al., 2011)

Hsap\HTT^{Q138.UAS.mRFP1}, Scer\GAL4^elav-C155 has lethal | pupal stage phenotype, suppressible by lkb1[+]/Lkb1^4A4-2

(Schulte et al., 2011)

Hsap\HTT^{Q138.UAS.mRFP1}, Scer\GAL4^elav-C155 has lethal | pupal stage phenotype, suppressible by Df(3L)vin7/+

(Schulte et al., 2011)

Hsap\HTT^{Q138.UAS.mRFP1}, Scer\GAL4^elav-C155 has abnormal locomotor rhythm phenotype, suppressible by lkb1[+]/Lkb1^4A4-2

(Schulte et al., 2011)

Other

Statement

Reference

Df(3L)vin7/+, Hsap\HTT^{Q138.UAS.mRFP1}, Scer\GAL4^elav-C155 has short lived phenotype

(Weiss et al., 2012)

Df(3L)vin7/+, Hsap\HTT^{Q138.UAS.mRFP1}, Scer\GAL4^elav-C155 has abnormal locomotor behavior | adult stage phenotype

(Weiss et al., 2012)

Df(2L)BSC109/+, Hsap\HTT^{Q138.UAS.mRFP1}, Scer\GAL4^elav-C155 has short lived phenotype

(Weiss et al., 2012)

Df(2L)BSC109/+, Hsap\HTT^{Q138.UAS.mRFP1}, Scer\GAL4^elav-C155 has abnormal locomotor behavior | adult stage phenotype

(Weiss et al., 2012)

Df(3R)Tpl10/+, Hsap\HTT^{Q138.UAS.mRFP1}, Scer\GAL4^elav-C155 has short lived phenotype

(Weiss et al., 2012)

Df(3R)Tpl10/+, Hsap\HTT^{Q138.UAS.mRFP1}, Scer\GAL4^elav-C155 has abnormal locomotor behavior | adult stage phenotype

(Weiss et al., 2012)

Df(3L)st-f13/+, Hsap\HTT^{Q138.UAS.mRFP1}, Scer\GAL4^elav-C155 has short lived phenotype

(Weiss et al., 2012)

Df(3L)st-f13/+, Hsap\HTT^{Q138.UAS.mRFP1}, Scer\GAL4^elav-C155 has abnormal locomotor behavior | adult stage phenotype

(Weiss et al., 2012)

+/Df(3L)brm11, Hsap\HTT^{Q138.UAS.mRFP1}, Scer\GAL4^elav-C155 has short lived phenotype

(Weiss et al., 2012)

+/Df(3L)brm11, Hsap\HTT^{Q138.UAS.mRFP1}, Scer\GAL4^elav-C155 has abnormal locomotor behavior | adult stage phenotype

(Weiss et al., 2012)

Df(3R)H-B79/+, Hsap\HTT^{Q138.UAS.mRFP1}, Scer\GAL4^elav-C155 has short lived phenotype

(Weiss et al., 2012)

Df(3R)H-B79/+, Hsap\HTT^{Q138.UAS.mRFP1}, Scer\GAL4^elav-C155 has abnormal locomotor behavior | adult stage phenotype

(Weiss et al., 2012)

Df(2L)JS17/+, Hsap\HTT^{Q138.UAS.mRFP1}, Scer\GAL4^elav-C155 has short lived phenotype

(Weiss et al., 2012)

Df(3L)BSC33/+, Hsap\HTT^{Q138.UAS.mRFP1}, Scer\GAL4^elav-C155 has short lived phenotype

(Weiss et al., 2012)

Df(3L)BSC33/+, Hsap\HTT^{Q138.UAS.mRFP1}, Scer\GAL4^elav-C155 has abnormal locomotor behavior | adult stage phenotype

(Weiss et al., 2012)

Df(2L)JS17/+, Hsap\HTT^{Q138.UAS.mRFP1}, Scer\GAL4^elav-C155 has abnormal locomotor behavior | adult stage phenotype

(Weiss et al., 2012)

Df(2R)cn9/+, Hsap\HTT^{Q138.UAS.mRFP1}, Scer\GAL4^elav-C155 has short lived phenotype

(Weiss et al., 2012)

Df(2R)cn9/+, Hsap\HTT^{Q138.UAS.mRFP1}, Scer\GAL4^elav-C155 has abnormal locomotor behavior | adult stage phenotype

(Weiss et al., 2012)

Df(2R)AA21/+, Hsap\HTT^{Q138.UAS.mRFP1}, Scer\GAL4^elav-C155 has short lived phenotype

(Weiss et al., 2012)

Df(2R)AA21/+, Hsap\HTT^{Q138.UAS.mRFP1}, Scer\GAL4^elav-C155 has abnormal locomotor behavior | adult stage phenotype

(Weiss et al., 2012)

+/Df(2R)59AD, Hsap\HTT^{Q138.UAS.mRFP1}, Scer\GAL4^elav-C155 has short lived phenotype

(Weiss et al., 2012)

+/Df(2R)59AD, Hsap\HTT^{Q138.UAS.mRFP1}, Scer\GAL4^elav-C155 has abnormal locomotor behavior | adult stage phenotype

(Weiss et al., 2012)

Phenotype Manifest In

NOT Enhanced by

Statement

Reference

Hsap\HTT^{Q138.UAS.mRFP1}, Scer\GAL4^Appl.G1a has mitochondrion | larval stage phenotype, non-enhanceable by Pi3K92E^CAAX.UAS, Scer\GAL4^Appl.G1a

(Swinter et al., 2023)

Hsap\HTT^{Q138.UAS.mRFP1}, Scer\GAL4^Appl.G1a has larval segmental nerve phenotype, non-enhanceable by Pi3K92E^CAAX.UAS, Scer\GAL4^Appl.G1a

(Swinter et al., 2023)

Suppressed by

Statement

Reference

Hsap\HTT^{Q138.UAS.mRFP1}, Scer\GAL4^Appl.G1a has mitochondrion | larval stage phenotype, suppressible by Hsap\HSPA1L^UAS.cWa, Scer\GAL4^Appl.G1a

(Swinter et al., 2023)

Hsap\HTT^{Q138.UAS.mRFP1}, Scer\GAL4^Appl.G1a has larval segmental nerve phenotype, suppressible by Hsap\HSPA1L^UAS.cWa, Scer\GAL4^Appl.G1a

(Swinter et al., 2023)

Hsap\HTT^{Q138.UAS.mRFP1}, Scer\GAL4^Appl.G1a has axon | larval stage phenotype, suppressible by Hsap\HSPA1L^UAS.cWa, Scer\GAL4^Appl.G1a

(Swinter et al., 2023)

NOT suppressed by

Statement

Reference

Hsap\HTT^{Q138.UAS.mRFP1}, Scer\GAL4^Appl.G1a has mitochondrion | larval stage phenotype, non-suppressible by Pi3K92E^CAAX.UAS, Scer\GAL4^Appl.G1a

(Swinter et al., 2023)

Hsap\HTT^{Q138.UAS.mRFP1}, Scer\GAL4^Appl.G1a has larval segmental nerve phenotype, non-suppressible by Pi3K92E^CAAX.UAS, Scer\GAL4^Appl.G1a

(Swinter et al., 2023)

Additional Comments

Genetic Interactions

Statement

Reference

Xenogenetic Interactions

Statement

Reference

One copy of Df(3L)vin7 fully rescues the lethality seen when Hsap\HTT^{Q138.Scer\UAS.T:Disc\RFP-mRFP} is expressed under the control of Scer\GAL4^elav-C155 using the P{UAS-HTT.Q138.mRFP}A insertion line. However the viable animals display motor defects, as they are unable to climb the walls of the vials or mate, and live to around 10 days post-eclosion.

One copy of Df(2L)JS17 partially rescues the lethality seen when Hsap\HTT^{Q138.Scer\UAS.T:Disc\RFP-mRFP} is expressed under the control of Scer\GAL4^elav-C155 using the P{UAS-HTT.Q138.mRFP}A insertion line. The viable animals display motor defects, as they are unable to climb the walls of the vials or mate, and most die within several days of eclosion.

One copy of Df(2R)cn9 partially rescues the lethality seen when Hsap\HTT^{Q138.Scer\UAS.T:Disc\RFP-mRFP} is expressed under the control of Scer\GAL4^elav-C155 using the P{UAS-HTT.Q138.mRFP}A insertion line. The viable animals display motor defects, as they are unable to climb the walls of the vials or mate, and most die within several days of eclosion.

One copy of Df(2R)AA21 partially rescues the lethality seen when Hsap\HTT^{Q138.Scer\UAS.T:Disc\RFP-mRFP} is expressed under the control of Scer\GAL4^elav-C155 using the P{UAS-HTT.Q138.mRFP}A insertion line. The viable animals display motor defects, as they are unable to climb the walls of the vials or mate, and most die within several days of eclosion.

One copy of Df(2R)59AD partially rescues the lethality seen when Hsap\HTT^{Q138.Scer\UAS.T:Disc\RFP-mRFP} is expressed under the control of Scer\GAL4^elav-C155 using the P{UAS-HTT.Q138.mRFP}A insertion line. The viable animals display motor defects, as they are unable to climb the walls of the vials or mate, and most die within several days of eclosion.

One copy of Df(2L)BSC109 partially rescues the lethality seen when Hsap\HTT^{Q138.Scer\UAS.T:Disc\RFP-mRFP} is expressed under the control of Scer\GAL4^elav-C155 using the P{UAS-HTT.Q138.mRFP}A insertion line. The viable animals display motor defects, as they are unable to climb the walls of the vials or mate, and most die within several days of eclosion.

One copy of Df(3R)Tpl10 partially rescues the lethality seen when Hsap\HTT^{Q138.Scer\UAS.T:Disc\RFP-mRFP} is expressed under the control of Scer\GAL4^elav-C155 using the P{UAS-HTT.Q138.mRFP}A insertion line. The viable animals display motor defects, as they are unable to climb the walls of the vials or mate, and most die within several days of eclosion.

One copy of Df(3L)st-f13 partially rescues the lethality seen when Hsap\HTT^{Q138.Scer\UAS.T:Disc\RFP-mRFP} is expressed under the control of Scer\GAL4^elav-C155 using the P{UAS-HTT.Q138.mRFP}A insertion line. The viable animals display motor defects, as they are unable to climb the walls of the vials or mate, and most die within several days of eclosion.

One copy of Df(3L)brm11 partially rescues the lethality seen when Hsap\HTT^{Q138.Scer\UAS.T:Disc\RFP-mRFP} is expressed under the control of Scer\GAL4^elav-C155 using the P{UAS-HTT.Q138.mRFP}A insertion line. The viable animals display motor defects, as they are unable to climb the walls of the vials or mate, and most die within several days of eclosion.

One copy of Df(3R)H-B79 partially rescues the lethality seen when Hsap\HTT^{Q138.Scer\UAS.T:Disc\RFP-mRFP} is expressed under the control of Scer\GAL4^elav-C155 using the P{UAS-HTT.Q138.mRFP}A insertion line. The viable animals display motor defects, as they are unable to climb the walls of the vials or mate, and most die within several days of eclosion.

One copy of Df(3L)BSC33 partially rescues the lethality seen when Hsap\HTT^{Q138.Scer\UAS.T:Disc\RFP-mRFP} is expressed under the control of Scer\GAL4^elav-C155 using the P{UAS-HTT.Q138.mRFP}A insertion line. The viable animals display motor defects, as they are unable to climb the walls of the vials or mate, and most die within several days of eclosion.

One copy of Df(2R)59AD suppresses the Hsap\HTT aggregation seen in salivary glands when Hsap\HTT^{Q138.Scer\UAS.T:Disc\RFP-mRFP} is expressed under the control of Scer\GAL4^elav-C155 using the lower-expression P{UAS-HTT.Q138.mRFP}B insertion line. The number of axonal aggregates is not reduced.

One copy of Df(2R)AA21 suppresses the Hsap\HTT aggregation seen in salivary glands when Hsap\HTT^{Q138.Scer\UAS.T:Disc\RFP-mRFP} is expressed under the control of Scer\GAL4^elav-C155 using the lower-expression P{UAS-HTT.Q138.mRFP}B insertion line. The number of axonal aggregates is not reduced.

One copy of Df(2R)59AB suppresses the Hsap\HTT aggregation seen in salivary glands when Hsap\HTT^{Q138.Scer\UAS.T:Disc\RFP-mRFP} is expressed under the control of Scer\GAL4^elav-C155 using the lower-expression P{UAS-HTT.Q138.mRFP}B insertion line. The number of axonal aggregates is not reduced.

One copy of Df(2R)59AB partially rescues the lethality seen when Hsap\HTT^{Q138.Scer\UAS.T:Disc\RFP-mRFP} is expressed under the control of Scer\GAL4^elav-C155 using the P{UAS-HTT.Q138.mRFP}A insertion line.

One copy of Df(2R)cn9 suppresses the Hsap\HTT aggregation seen in salivary glands when Hsap\HTT^{Q138.Scer\UAS.T:Disc\RFP-mRFP} is expressed under the control of Scer\GAL4^elav-C155 using the lower-expression P{UAS-HTT.Q138.mRFP}B insertion line. The number of axonal aggregates is not reduced.

One copy of Df(2R)sple-J1 suppresses the Hsap\HTT aggregation seen in salivary glands when Hsap\HTT^{Q138.Scer\UAS.T:Disc\RFP-mRFP} is expressed under the control of Scer\GAL4^elav-C155 using the lower-expression P{UAS-HTT.Q138.mRFP}B insertion line. The number of axonal aggregates is not reduced.

One copy of Df(2R)sple-J1 partially rescues the lethality seen when Hsap\HTT^{Q138.Scer\UAS.T:Disc\RFP-mRFP} is expressed under the control of Scer\GAL4^elav-C155 using the P{UAS-HTT.Q138.mRFP}A insertion line.

One copy of Df(3L)vin7 suppresses the Hsap\HTT aggregation seen in salivary glands when Hsap\HTT^{Q138.Scer\UAS.T:Disc\RFP-mRFP} is expressed under the control of Scer\GAL4^elav-C155 using the lower-expression P{UAS-HTT.Q138.mRFP}B insertion line. The number of axonal aggregates is also reduced.

One copy of Df(3L)ED220 significantly rescues the lethality seen when Hsap\HTT^{Q138.Scer\UAS.T:Disc\RFP-mRFP} is expressed under the control of Scer\GAL4^elav-C155 using the P{UAS-HTT.Q138.mRFP}A insertion line.

One copy of Df(3R)MAP117 significantly rescues the lethality seen when Hsap\HTT^{Q138.Scer\UAS.T:Disc\RFP-mRFP} is expressed under the control of Scer\GAL4^elav-C155 using the P{UAS-HTT.Q138.mRFP}A insertion line.

One copy of Df(3R)MAP2 significantly rescues the lethality seen when Hsap\HTT^{Q138.Scer\UAS.T:Disc\RFP-mRFP} is expressed under the control of Scer\GAL4^elav-C155 using the P{UAS-HTT.Q138.mRFP}A insertion line.

One copy of CG5830^c02324 rescues the lethality seen when Hsap\HTT^{Q138.Scer\UAS.T:Disc\RFP-mRFP} is expressed under the control of Scer\GAL4^elav-C155 using the P{UAS-HTT.Q138.mRFP}A insertion line. The number of axonal aggregates seen in the peripheral nerves of third instar larvae is not reduced.

One copy of lab¹⁴ significantly rescues the lethality seen when Hsap\HTT^{Q138.Scer\UAS.T:Disc\RFP-mRFP} is expressed under the control of Scer\GAL4^elav-C155 using the P{UAS-HTT.Q138.mRFP}A insertion line. The number of axonal aggregates seen in the peripheral nerves of third instar larvae is not reduced.

One copy of Scr^CP1 partially rescues the lethality seen when Hsap\HTT^{Q138.Scer\UAS.T:Disc\RFP-mRFP} is expressed under the control of Scer\GAL4^elav-C155 using the P{UAS-HTT.Q138.mRFP}A insertion line.

One copy of Scr⁶ partially rescues the lethality seen when Hsap\HTT^{Q138.Scer\UAS.T:Disc\RFP-mRFP} is expressed under the control of Scer\GAL4^elav-C155 using the P{UAS-HTT.Q138.mRFP}A insertion line.

One copy of pb¹ partially rescues the lethality seen when Hsap\HTT^{Q138.Scer\UAS.T:Disc\RFP-mRFP} is expressed under the control of Scer\GAL4^elav-C155 using the P{UAS-HTT.Q138.mRFP}A insertion line.

One copy of Dfd⁶ partially rescues the lethality seen when Hsap\HTT^{Q138.Scer\UAS.T:Disc\RFP-mRFP} is expressed under the control of Scer\GAL4^elav-C155 using the P{UAS-HTT.Q138.mRFP}A insertion line.

One copy of Ubx⁵¹ partially rescues the lethality seen when Hsap\HTT^{Q138.Scer\UAS.T:Disc\RFP-mRFP} is expressed under the control of Scer\GAL4^elav-C155 using the P{UAS-HTT.Q138.mRFP}A insertion line.

(Weiss et al., 2012)

lkb1^4B1-11 or lkb1^4A4-2 heterozygous animals expressing Hsap\HTT^{Q138.Scer\UAS.T:Disc\RFP-mRFP} pan-neuronally (under the control of Scer\GAL4^elav-C155) exhibit an adult escaper frequency of 1.8% and 3.7% respectively. These animals are viable and have a relatively normal walking ability, although they do not inflate their wings.

The introduction of an lkb1^4A4-2 heterozygous background enhances the climbing ability of flies expressing Hsap\HTT^{Q138.Scer\UAS.T:Disc\RFP-mRFP} under the control of Scer\GAL4^elav-C155. However, this has no effect on controls.

(Schulte et al., 2011)

Complementation and Rescue Data

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Hsap\HTT^{Q138.Scer\UAS.T:Disc\RFP-mRFP}

Hsap\HTT^{Q138.UAS.mRFP1}

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