FlyBase Allele Report: Dmel\Trpml[1]

General Information

Symbol

Dmel\Trpml¹

Species

D. melanogaster

Name

FlyBase ID

FBal0221782

Feature type

allele

Associated gene

Dmel\Trpml

Associated Insertion(s)

Carried in Construct

Key Links

Genomic Maps

JBrowse

Allele class

amorphic allele - genetic evidence

Mutagen

P-element activity

Nature of the Allele

Allele class

amorphic allele - genetic evidence

(Venkatachalam et al., 2008)

Mutagen

P-element activity

(Venkatachalam et al., 2008)

Progenitor genotype

P{EP}Trpml^GE22279

(Venkatachalam et al., 2008)

Cytology

Description

Imprecise excision of the progenitor insertion, resulting in a 1.1kb deletion which removes nucleotides -456 to +641 relative to the trpml translation start site.

(Venkatachalam et al., 2008)

Mutations Mapped to the Genome

Curation Data

Type

Location

Additional Notes

References

deletion

3L:19,717,427..19,718,524 [-]

Comment:

Imprecise excision of the progenitor insertion, resulting in a 1.1kb deletion which removes nucleotides -456 to +641 relative to the Trpml translation start site.

(Venkatachalam et al., 2008)

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 glycoproteinosis

CEA

(Venkatachalam et al., 2008)

model of mucolipidosis type IV

CEA

(Wong et al., 2015)

CEA with Trpml²

(Wong et al., 2015)

Modifiers Based on Experimental Evidence ( 1 )

Disease

Interaction

References

model of glycoproteinosis

is ameliorated by Hsap\HSPA1L^UAS.cWa

(Venkatachalam et al., 2008)

exacerbates neurodegenerative disease

modeled by Hsap\HTT^GMR.Q120

(Venkatachalam et al., 2008)

model of mucolipidosis type IV

is ameliorated by RagA-B^Q61L.UAS

(Wong et al., 2015)

is exacerbated by bsk¹

(Wong et al., 2015)

is exacerbated by hiw^UAS.fl

(Wong et al., 2015)

is exacerbated by wnd³

(Wong et al., 2015)

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

Disease-implicated variant(s)

Phenotypic Data

Phenotypic Class

abnormal developmental rate | pupal stage

(Wong et al., 2012)

abnormal immune response | adult stage | recessive

(Wong et al., 2017)

abnormal locomotor behavior | adult stage

(Venkatachalam et al., 2008)

abnormal locomotor behavior | adult stage (with Df(3L)ED228)

(Venkatachalam et al., 2008)

abnormal locomotor behavior | adult stage (with Df(3L)Exel6135)

(Venkatachalam et al., 2008)

abnormal neuroanatomy | third instar larval stage

(Wong et al., 2015)

abnormal neuroanatomy | third instar larval stage (with Df(3L)Exel6135)

(Wong et al., 2015)

abnormal neuroanatomy | third instar larval stage (with Trpml²)

(Wong et al., 2015)

abnormal neurophysiology | third instar larval stage

(Venkatachalam et al., 2008)

increased cell death

(Venkatachalam et al., 2008)

lethal (with Df(3L)Exel6135)

(Sandstrom, 2011)

lethal - all die before end of pupal stage

(Feng et al., 2014)

lethal - all die before end of pupal stage (with Df(3L)ED228)

(Venkatachalam et al., 2008)

lethal - all die before end of pupal stage (with Df(3L)Exel6135)

(Venkatachalam et al., 2008)

lethal - all die before end of pupal stage (with Trpml²)

(Venkatachalam et al., 2008)

lethal - all die before end of pupal stage | recessive

(Venkatachalam et al., 2008)

partially lethal - majority die

(Wong et al., 2015, Wong et al., 2012)

partially lethal - majority die (with Trpml^c05523)

(Sandstrom, 2011)

some die during pharate adult stage

(Wong et al., 2015)

some die during pupal stage

(Wong et al., 2012)

some die during pupal stage (with Df(3L)ED228)

(Venkatachalam et al., 2008)

some die during pupal stage (with Df(3L)Exel6135)

(Venkatachalam et al., 2008)

some die during pupal stage (with Trpml²)

(Venkatachalam et al., 2008)

some die during pupal stage | recessive

(Venkatachalam et al., 2008)

Phenotype Manifest In

adult brain

(Venkatachalam et al., 2008)

amphisome

(Wong et al., 2012)

autophagosome

(Wong et al., 2012)

embryonic/larval fat body | second instar larval stage

(Wong et al., 2012)

embryonic/larval fat body | third instar larval stage

(Wong et al., 2012)

embryonic/larval neuromuscular junction

(Venkatachalam et al., 2008)

embryonic/larval neuromuscular junction | third instar larval stage

(Wong et al., 2015)

embryonic/larval neuromuscular junction | third instar larval stage (with Df(3L)Exel6135)

(Wong et al., 2015)

embryonic/larval neuromuscular junction | third instar larval stage (with Trpml²)

(Wong et al., 2015)

eye photoreceptor cell

(Venkatachalam et al., 2008)

lysosome

(Wong et al., 2012)

mitochondrion

(Venkatachalam et al., 2008)

multivesicular body

(Wong et al., 2012)

NMJ bouton | third instar larval stage

(Wong et al., 2015)

NMJ bouton | third instar larval stage (with Df(3L)Exel6135)

(Wong et al., 2015)

NMJ bouton | third instar larval stage (with Trpml²)

(Wong et al., 2015)

photoreceptor neuron | adult stage

(Wong et al., 2012)

rhabdomere

(Venkatachalam et al., 2008)

wing (with Trpml^c05523)

(Sandstrom, 2011)

Detailed Description

Statement

Reference

Mature eggs from Trpml¹ homozygous females show similar incidence of calcium waves as heterozygous controls upon in vitro egg activation.

(Hu and Wolfner, 2019)

Trpml¹ individuals are unable to clear bacteria after injection of the non-pathogenic bacteria, E. coli, as compared to equally infected heterozygous controls.

(Wong et al., 2017)

Trpml¹ mutant third instar larvae show dramatic enlargement of lysosomes (marked by Lysotracker) in the somatic muscles.

(Bharadwaj et al., 2016)

Trpml¹ mutant adults show normal preference for less solid sucrose-agarose food than a harder one (higher agarose conc., same sucrose content) one in a two-way food choice assay.

(Zhang et al., 2016)

Trpml¹/Trpml¹ third instar larvae display fewer neuromuscular junction boutons compared to wild-type controls, but the bouton morphology remains normal.

Trpml¹/Trpml² transheterozygous third instar larvae display fewer neuromuscular junction boutons compared to wild-type controls.

Trpml¹/Df(3L)Exel6135 third instar larvae display fewer neuromuscular junction boutons compared to wild-type controls.

Supplementing food with ML-SA1 does not restore normal synaptic growth (number of neuromuscular junction boutons) in Trpml¹ homozygous larvae.

Raising Trpml¹ mutant larvae on a protein-rich diet does not alleviate the synaptic growth defects and the number of neuromuscular junction boutons in Trpml¹ third instar larvae remains decreased compared to wild-type controls.

Raising Trpml¹ mutant larvae on a protein-rich diet supplemented with the ALK inhibitor CH5424802 alleviates the synaptic growth defects and the decreased number of neuromuscular junction boutons characteristic for Trpml¹ third instar larvae is restored compared to controls.

Raising Trpml¹ mutants on a protein-rich diet supplemented with the ALK inhibitor CH5424802 increases the number of adults eclosing from pupal cases compared to Trpml¹ mutant controls raised on protein-rich diet alone.

(Wong et al., 2015)

The gustatory aversion of mutant flies to 6mM camphor in a two way-choice test is not significantly different from that seen in wild-type flies.

(Zhang et al., 2013)

trpml¹ mutant fat body cells from wandering third instar larvae contain an increased number of autophagosomes and amphisomes compared to controls. The fat body cells also contain fewer and smaller lysosomes. The cells accumulate large single-membrane bound multivesicular bodies (MVBs)/amphisomes, many of which are touching lysosomes, apparently unable to go fusion; these are referred to as "fusion clamped" vesicles. Pairs of "fusion clamped" lysosomes are also seen.

The volume of LysoTracker-positive vesicles is increased in trpml¹ mutant larval fat body cells. The increase is most pronounced in second instar larvae, but is still significant at the third instar stage. This phenotype is suppressed when the larvae are fed protein-rich yeast paste, or if the fat body is treated with thapsigargin, which blocks the sarcoplasmic/endoplasmic reticulum calcium ATPase pump.

trpml¹ mutant adult photoreceptor cells show a dramatic elevation in the number of both multivesicular bodies (MVBs) and lysosomes, as well as accumulation of autophagosomes. The number of amphisomes is also increased. Despite these increases, the number of autolysosomes is not significantly different from wild type and the ratio of MVBs to autolysosomes is 10 fold higher in trpml¹ mutant cells compared to controls. The number of MVBs touching lysosomes ("fusion-clamped vesicles") is significantly higher than in wild type.

The half-maximal time to pupation is increased compared to controls in trpml¹ mutants, and this phenotype is rescued when the larvae are fed protein-rich yeast paste.

Less than 10% of trpml¹ mutants eclose from the pupal cases. This lethality is significantly rescued when the flies are fed a high-protein diet. Lethality is also rescued when the larvae are fed food that has been supplemented with tryptone. Supplementation with sucrose has no effect on lethality. Feeding the larvae the TORC1 inhibitor Rapamycin enhances lethality when flies are reared on normal food, and suppresses the rescuing effect seen when flies are fed high-protein yeast paste.

(Wong et al., 2012)

trpml^c05523/trpml¹ adult escapers have a 100% penetrant crumpled wing phenotype.

trpml¹/Df(3L)Exel6135 adult escapers show normal baseline excitatory junctional current amplitudes at the neuromuscular junction at both pH 5.75 and under reduced Mg[2+] conditions.

(Sandstrom, 2011)

Mutant adults show normal avoidance of 1% citronellal in a direct airborne repellent test (DART) assay.

(Kwon et al., 2010)

trpml¹ mutants retain a preference for 18[o]C (i.e. are thermotactic).

(Kwon et al., 2008)

5 day old trpml¹ adult escapers show a significantly reduced locomotor activity compared to controls. Homozygous 5 day old adult escapers show reduced climbing ability compared to controls in a negative geotaxis assay, and by 21 days after eclosion, negative geotaxis is nearly eliminated in the mutant flies.

21 day old trpml¹/Df(3L)ED228 and trpml¹/Df(3L)Exel6135 adult escapers show almost complete elimination of climbing ability in a negative geotaxis assay.

Ommatidia of newly eclosed trpml¹ escapers contain the full set of rhabdomeres. However, a significantly lower fraction of ommatidia (53.8 +/- 9.9%) retain all seven rhabdomeres in 21 day old trpml¹ flies compared to control flies of the same age (100% of ommatidia have 7 rhabdomeres in controls). The loss of rhabdomeres seen the trpml¹ flies is partially suppressed if the flies are reared in the dark.

The brains of young trpml¹ adults show minimal TUNEL staining, but the brains of 21 day old trpml¹ adults show increased TUNEL staining. The brains of 21 day old trpml¹ adults show a significant elevation in the accumulation of both early apoptotic cells and late apoptotic/necrotic cells compared to controls.

Photoreceptor cell bodies of 21 day old trpml¹ adults have 6.5-fold more cytoplasmic inclusions than wild type. The inclusions vary in diameter from 0.2-1.0μm and consist of multilamellar and multivesicular bodies. This accumulation of cytoplasmic inclusions in the mutant flies is age dependent.

There is a small but significant increase in the number of mitochondria in the photoreceptor cell bodies of 21 day old trpml¹ adults. However, there is a 4-fold decrease in the intensity of Mitotracker-orange GM-H2TMRos staining, indicating accumulation of damaged mitochondria.

21 day old trpml¹ adults have approximately 40% higher hydrogen peroxide levels compared to controls.

trpml¹ adults remain immobilised longer than wild type after exposure to CO[[2]] anesthesia.

The amplitude of the excitatory junctional potential (EJP) at the neuromuscular junction is decreased approximately 50% in trpml¹ third instar larvae compared to controls.

(Venkatachalam et al., 2008)

External Data

Linkouts

Interactions

Show genetic interaction network for Enhancers & Suppressors

Phenotypic Class

NOT Enhanced by

Statement

Reference

Trpml¹ has abnormal neuroanatomy | third instar larval stage phenotype, non-enhanceable by Scer\GAL4^elav-C155/RagA-B^T16N.UAS

(Wong et al., 2015)

Trpml¹ has abnormal neuroanatomy | third instar larval stage phenotype, non-enhanceable by Scer\GAL4^elav-C155/AMPKα^T184D.UAS

(Wong et al., 2015)

Suppressed by

Statement

Reference

Trpml¹ has partially lethal - majority die phenotype, suppressible by Scer\GAL4^elav-C155/RagA-B^Q61L.UAS

(Wong et al., 2015)

Trpml¹ has some die during pharate adult stage phenotype, suppressible by Scer\GAL4^elav-C155/RagA-B^Q61L.UAS

(Wong et al., 2015)

Trpml¹ has abnormal neuroanatomy | third instar larval stage phenotype, suppressible by Scer\GAL4^elav-C155/RagA-B^Q61L.UAS

(Wong et al., 2015)

Trpml¹ has abnormal neuroanatomy | third instar larval stage phenotype, suppressible by Scer\GAL4^nSyb.PS/RagA-B^Q61L.UAS

(Wong et al., 2015)

Trpml¹ has lethal - all die before end of pupal stage phenotype, suppressible by Scer\GAL4^elav-C155/Hsap\MCOLN1^UAS.Tag:HA

(Feng et al., 2014)

Trpml¹ has lethal phenotype, suppressible by Scer\GAL4^elav-C155/crq^UAS.cFa

(Venkatachalam et al., 2008)

Trpml¹ has lethal phenotype, suppressible by Scer\GAL4^Hml.PG/crq^UAS.cFa

(Venkatachalam et al., 2008)

Trpml¹ has abnormal locomotor behavior | adult stage phenotype, suppressible by Scer\GAL4^elav-C155/crq^UAS.cFa

(Venkatachalam et al., 2008)

Trpml¹ has lethal phenotype, suppressible by Scer\GAL4^Act5C.PI/Hsap\HSPA1L^UAS.cWa

(Venkatachalam et al., 2008)

Trpml¹ has lethal phenotype, suppressible by Scer\GAL4^elav-C155/Hsap\HSPA1L^UAS.cWa

(Venkatachalam et al., 2008)

Trpml¹ has abnormal locomotor behavior | adult stage phenotype, suppressible by Scer\GAL4^Act5C.PI/Hsap\HSPA1L^UAS.cWa

(Venkatachalam et al., 2008)

Trpml¹ has abnormal locomotor behavior | adult stage phenotype, suppressible by Scer\GAL4^elav-C155/Hsap\HSPA1L^UAS.cWa

(Venkatachalam et al., 2008)

NOT suppressed by

Statement

Reference

Trpml¹ has abnormal neuroanatomy | third instar larval stage phenotype, non-suppressible by Scer\GAL4^elav-C155/AMPKα^K56R.UAS

(Wong et al., 2015)

Trpml¹ has lethal phenotype, non-suppressible by Scer\GAL4^repo/Hsap\HSPA1L^UAS.cWa

(Venkatachalam et al., 2008)

Trpml¹ has lethal phenotype, non-suppressible by Scer\GAL4^Cg.PA/Hsap\HSPA1L^UAS.cWa

(Venkatachalam et al., 2008)

Trpml¹ has lethal phenotype, non-suppressible by Scer\GAL4^c754/Hsap\HSPA1L^UAS.cWa

(Venkatachalam et al., 2008)

Suppressor of

Statement

Reference

Trpml¹/Trpml[+] is a suppressor of abnormal immune response phenotype of ClC-b^UAS.cWa, Scer\GAL4^He.PZ

(Wong et al., 2017)

Trpml¹ is a suppressor of lethal - all die during P-stage | recessive phenotype of AMPKα¹

(Onyenwoke et al., 2015)

Trpml¹ is a suppressor of lethal - all die during P-stage | recessive phenotype of AMPKα³

(Onyenwoke et al., 2015)

Trpml¹ is a suppressor | partially of lethal - all die during P-stage phenotype of AMPKα^JF01951, Scer\GAL4^Act.PU

(Onyenwoke et al., 2015)

Trpml¹ is a suppressor of abnormal neuroanatomy | third instar larval stage phenotype of hiw^ND8

(Wong et al., 2015)

Other

Statement

Reference

Scer\GAL4^elav-C155, Trpml¹, hiw^UAS.fl has abnormal neuroanatomy | third instar larval stage phenotype

(Wong et al., 2015)

cln3[+]/Cln3^ΔMB1, Trpml¹ has abnormal neuroanatomy | third instar larval stage phenotype

(Wong et al., 2015)

Scer\GAL4^elav-C155, Trpml¹, hiw^UAS.fl has lethal - all die during embryonic stage | recessive phenotype

(Wong et al., 2015)

Trpml¹, bsk¹/bsk[+] has abnormal neuroanatomy | third instar larval stage phenotype

(Wong et al., 2015)

Trpml¹, wnd[+]/wnd³ has abnormal neuroanatomy | third instar larval stage phenotype

(Wong et al., 2015)

Thor², Trpml¹ has lethal | pupal stage phenotype

(Wong et al., 2012)

Phenotype Manifest In

NOT Enhanced by

Statement

Reference

Trpml¹ has neuromuscular junction | third instar larval stage phenotype, non-enhanceable by Scer\GAL4^elav-C155/RagA-B^T16N.UAS

(Wong et al., 2015)

Trpml¹ has NMJ bouton | third instar larval stage phenotype, non-enhanceable by Scer\GAL4^elav-C155/RagA-B^T16N.UAS

(Wong et al., 2015)

Trpml¹ has neuromuscular junction | third instar larval stage phenotype, non-enhanceable by Scer\GAL4^elav-C155/AMPKα^T184D.UAS

(Wong et al., 2015)

Trpml¹ has NMJ bouton | third instar larval stage phenotype, non-enhanceable by Scer\GAL4^elav-C155/AMPKα^T184D.UAS

(Wong et al., 2015)

Suppressed by

Statement

Reference

Trpml¹ has neuromuscular junction | third instar larval stage phenotype, suppressible by Scer\GAL4^elav-C155/RagA-B^Q61L.UAS

(Wong et al., 2015)

Trpml¹ has NMJ bouton | third instar larval stage phenotype, suppressible by Scer\GAL4^elav-C155/RagA-B^Q61L.UAS

(Wong et al., 2015)

Trpml¹ has neuromuscular junction | third instar larval stage phenotype, suppressible by Scer\GAL4^nSyb.PS/RagA-B^Q61L.UAS

(Wong et al., 2015)

Trpml¹ has NMJ bouton | third instar larval stage phenotype, suppressible by Scer\GAL4^nSyb.PS/RagA-B^Q61L.UAS

(Wong et al., 2015)

Trpml¹ has embryonic/larval fat body | second instar larval stage phenotype, suppressible by Rheb^UAS.cUa/Scer\GAL4^Cg.PA

(Wong et al., 2012)

Trpml¹ has embryonic/larval fat body | third instar larval stage phenotype, suppressible by Rheb^UAS.cUa/Scer\GAL4^Cg.PA

(Wong et al., 2012)

NOT suppressed by

Statement

Reference

Trpml¹ has neuromuscular junction | third instar larval stage phenotype, non-suppressible by Scer\GAL4^elav-C155/AMPKα^K56R.UAS

(Wong et al., 2015)

Trpml¹ has NMJ bouton | third instar larval stage phenotype, non-suppressible by Scer\GAL4^elav-C155/AMPKα^K56R.UAS

(Wong et al., 2015)

Enhancer of

Statement

Reference

Trpml¹ is an enhancer of eye photoreceptor cell phenotype of Hsap\HTT^GMR.Q120

(Venkatachalam et al., 2008)

Suppressor of

Statement

Reference

Trpml¹ is a suppressor | partially of larval glial cell | larval stage phenotype of Hsap\HRAS^{G12V.UAS.GFP,Tag:HA}, Scer\GAL4^repo

(Jung et al., 2019)

Trpml¹ is a suppressor of NMJ bouton | third instar larval stage phenotype of hiw^ND8

(Wong et al., 2015)

Trpml¹ is a suppressor of embryonic/larval neuromuscular junction | third instar larval stage phenotype of hiw^ND8

(Wong et al., 2015)

Other

Statement

Reference

Trpml¹, wnd[+]/wnd³ has NMJ bouton | third instar larval stage phenotype

(Wong et al., 2015)

cln3[+]/Cln3^ΔMB1, Trpml¹ has neuromuscular junction | third instar larval stage phenotype

(Wong et al., 2015)

cln3[+]/Cln3^ΔMB1, Trpml¹ has NMJ bouton | third instar larval stage phenotype

(Wong et al., 2015)

Scer\GAL4^elav-C155, Trpml¹, hiw^UAS.fl has neuromuscular junction | third instar larval stage phenotype

(Wong et al., 2015)

Scer\GAL4^elav-C155, Trpml¹, hiw^UAS.fl has NMJ bouton | third instar larval stage phenotype

(Wong et al., 2015)

Trpml¹, bsk¹/bsk[+] has neuromuscular junction | third instar larval stage phenotype

(Wong et al., 2015)

Trpml¹, bsk¹/bsk[+] has NMJ bouton | third instar larval stage phenotype

(Wong et al., 2015)

Trpml¹, wnd[+]/wnd³ has neuromuscular junction | third instar larval stage phenotype

(Wong et al., 2015)

Additional Comments

Genetic Interactions

Statement

Reference

The late pupal lethality observed in animals expressing AMPKα^JF01951 under the control of Scer\GAL4^Act.PU can be partially rescued by combination with either Trpml¹ or Trpml².

The pupal-stage lethality of either hemizygous AMPKα¹ or AMPKα³ males can be partially rescued by combination with either Trpml¹ or Trpml².

(Onyenwoke et al., 2015)

Ectopic expression of RagA-B^{T16N.Scer\UAS} under the control of the Scer\GAL4^elav-C155 driver does not cause further decrease in the number of neuromuscular junction (NMJ) boutons in the already NMJ boutons-deficient Trpml¹ mutant third instar larvae.

Ectopic expression of RagA-B^{Q61L.Scer\UAS} under the control of the Scer\GAL4^elav-C155 or Scer\GAL4^nSyb.PS driver fully restores the decreased number of neuromuscular junction boutons characteristic for the Trpml¹ mutant third instar larvae.

Ectopic expression of RagA-B^{Q61L.Scer\UAS} under the control of the Scer\GAL4^elav-C155 driver in the Trpml¹ mutant background significantly increases the proportion of adults that eclose from pupal cases compared to the Trpml¹ mutants.

Ectopic expression of AMPKα^{T184D.Scer\UAS} under the control of the Scer\GAL4^elav-C155 driver does not cause further decrease in the number of neuromuscular junction (NMJ) boutons in the already NMJ boutons-deficient Trpml¹ mutant third instar larvae.

Ectopic expression of AMPKα^{K56R.Scer\UAS} under the control of the Scer\GAL4^elav-C155 driver does not restore the decreased number of neuromuscular junction boutons characteristic for the Trpml¹ mutant third instar larvae.

Trpml¹/+,bsk¹/+ double heterozygotes show decreased number of NMJ boutons in third instar larvae compared to either Trpml¹/+ or bsk¹/+ single heterozygotes.

Trpml¹/+;wnd³/+ double heterozygotes show decreased number of NMJ boutons in third instar larvae compared to either Trpml¹/+ or wnd³/+ single heterozygotes.

Ectopic expression of hiw^Scer\UAS.fl under the Scer\GAL4^elav-C155 driver in the Trpml¹/+ heterozygous background causes a decrease in the number of neuromuscular junction boutons in third instar larvae compared to controls.

Supplementing food with ML-SA1 (ML-synthetic agonist) does restore normal synaptic growth (number of neuromuscular junction boutons) in third instar Trpml¹/+ heterozygous larvae expressing hiw^Scer\UAS.fl under the Scer\GAL4^elav-C155 driver.

hiw^ND8;Trpml¹ double mutant larvae do not show the increase in number of neuromuscular junction boutons characteristic for the hiw^ND8 single mutants.

Trpml¹/+,cln3^ΔMB1/+ double heterozygotes show decreased number of NMJ boutons in third instar larvae compared to either Trpml¹/+ or cln3^ΔMB1/+ single heterozygotes.

Expression of RagA-B^{T16N.Scer\UAS} under the control of the Scer\GAL4^elav-C155 driver in Trpml¹ mutant larvae raised on a protein-rich diet supplemented with the ALK inhibitor CH5424802 precludes the alleviation of the synaptic growth defects and the decreased number of neuromuscular junction boutons characteristic for Trpml¹ third instar larvae remains low.

(Wong et al., 2015)

Expression of RagA-B^{Q61L.Scer\UAS} under the control of Scer\GAL4^Cg.PA suppresses the increase in Lysotracker-positive vesicle volume seen in trpml¹ mutant fat body cells.

Expression of Rheb^Scer\UAS.cUa under the control of Scer\GAL4^Cg.PA suppresses the increase in Lysotracker-positive vesicle volume seen in trpml¹ mutant fat body cells.

Expression of RagA-B^{T16N.Scer\UAS} under the control of Scer\GAL4^Cg.PA does not enhance the increase in Lysotracker-positive vesicle volume seen in trpml¹ mutant fat body cells.

Feeding larvae high-protein yeast paste does not suppress the pupal lethality seen in Thor² trpml¹ double mutants. The addition of Rapamycin also fails to suppress the lethality.

(Wong et al., 2012)

Xenogenetic Interactions

Statement

Reference

trpml¹ enhances the rate of age-dependent degeneration of photoreceptors that is caused by expression of Hsap\HD^GMR.Q120.

The lethality of trpml¹ homozygotes and the locomotor defects of trpml¹ homozygous adult escapers is suppressed by expression of Hsap\HSPA1L^Scer\UAS.cWa under the control of either Scer\GAL4^Act5C.PI or Scer\GAL4^elav-C155.

(Venkatachalam et al., 2008)

Complementation and Rescue Data

Rescued by

Trpml¹ is rescued by Trpml^UAS.cVa/Scer\GAL4^C164

(Wong et al., 2015)

Trpml¹/Trpml^c05523 is rescued by Trpml^+tVa

(Sandstrom, 2011)

Trpml¹/Df(3L)ED228 is rescued by Trpml^+tVa

(Venkatachalam et al., 2008)

Trpml¹ is rescued by Trpml^UAS.cVa/Scer\GAL4^Act5C.PI

(Venkatachalam et al., 2008)

Trpml¹/Trpml² is rescued by Trpml^+tVa

(Venkatachalam et al., 2008)

Trpml¹/Df(3L)Exel6135 is rescued by Trpml^+tVa

(Venkatachalam et al., 2008)

Partially rescued by

Trpml¹ is partially rescued by Trpml^UAS.cVa/Scer\GAL4^repo

(Venkatachalam et al., 2008)

Trpml¹ is partially rescued by Trpml^UAS.cVa/Scer\GAL4^Cg.PA

(Venkatachalam et al., 2008)

Trpml¹ is partially rescued by Trpml^UAS.cVa/Scer\GAL4^c754

(Venkatachalam et al., 2008)

Trpml¹ is partially rescued by Trpml^UAS.cVa/Scer\GAL4^Hml.PG

(Venkatachalam et al., 2008)

Not rescued by

Trpml¹ is not rescued by Trpml^UAS.cVa/Scer\GAL4^Mef2.PR

(Wong et al., 2015)

Trpml¹ is not rescued by Trpml^UAS.cVa/Scer\GAL4^e22c

(Venkatachalam et al., 2008)

Comments

The reduction in the number of neuromuscular junction boutons characteristic for Trpml¹ mutant third instar larvae is fully rescued by combination with Trpml^+tVa.

(Wong et al., 2015)

The crumpled wing phenotype seen in trpml^c05523/trpml¹ adult escapers is completely rescued by trpml^+tVa.

(Sandstrom, 2011)

Images (0)

Mutant

Wild-type

Stocks (4)

Bloomington

28992

w¹¹¹⁸; Trpml¹

27892

w¹¹¹⁸; P{Trpml^+tVa}2/CyO; Trpml¹

57372

w^*; P{UAS-Trpml.MYC}3, Trpml¹/TM6B, Tb¹

57373

w^*; P{UAS-MCOLN1.HA}2; Trpml¹/TM6B, Tb¹

Notes on Origin

Discoverer

External Crossreferences and Linkouts ( 0 )

Synonyms and Secondary IDs (3)

Reported As

Symbol Synonym

MCOLN¹

(Onyenwoke et al., 2015)

Trpml¹

trpml¹

(Tian et al., 2022, Ma and Freeman, 2020, Hu and Wolfner, 2019, Jung et al., 2019, Wong et al., 2017, Bharadwaj et al., 2016, Zhang et al., 2016, Onyenwoke et al., 2015, Wong et al., 2015, Feng et al., 2014, Zhang et al., 2013, Wong et al., 2012, Sandstrom, 2011, Cooper et al., 2010, Kwon et al., 2010, Kwon et al., 2010, Kwon et al., 2008, Venkatachalam et al., 2008)

Name Synonyms

Secondary FlyBase IDs

References (19)

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