FlyBase Allele Report: Dmel\wupA[hdp-3]

General Information

Symbol

Dmel\wupA^hdp-3

Species

D. melanogaster

Name

FlyBase ID

FBal0036021

Feature type

allele

Associated gene

Dmel\wupA

Associated Insertion(s)

Carried in Construct

Also Known As

hdp³, wupA^hdp3, heldup³

Key Links

Genomic Maps

JBrowse

Allele class

hypomorphic allele - genetic evidence

loss of function allele

Mutagen

ethyl methanesulfonate

Nature of the Allele

Allele class

hypomorphic allele - genetic evidence

(de la Pompa, 1994)

loss of function allele

(Barbas et al., 1993)

Mutagen

ethyl methanesulfonate

(Barbas et al., 1991, Deak et al., 1982)

Progenitor genotype

Cytology

Description

Splice junction defect.

(Barthmaier and Fyrberg, 1995)

One nucleotide displacement of the 3' AG splice site at the intron preceding exon 6b1, resulting in the failure to produce all exon 6b1-containing isoforms.

(Barbas et al., 1993)

Mutations Mapped to the Genome

Curation Data

Type

Location

Additional Notes

References

point_mutation

X:18,109,571..18,109,571 [-]

Nucleotide change:

G18109571A

Reported nucleotide change:

G?A

Comment:

One nucleotide displacement of the 3' AG splice site at the intron preceding exon 6b1, resulting in the failure to produce all exon 6b1-containing isoforms.

(Barbas et al., 1993)

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 ( 0 )

Disease

Evidence

References

Modifiers Based on Experimental Evidence ( 0 )

Disease

Interaction

References

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

Disease-implicated variant(s)

Phenotypic Data

Phenotypic Class

Phenotype Manifest In

coxal tergal remotor muscle cell

(Barbas et al., 1993)

dorsal longitudinal indirect flight muscle cell

(Nikonova et al., 2022, Nongthomba et al., 2004, Barbas et al., 1993)

indirect flight muscle & myofibril

(Nongthomba et al., 2004)

indirect flight muscle & myotube

(Nongthomba et al., 2004)

indirect flight muscle & sarcomere

(Nongthomba et al., 2004)

indirect flight muscle & striated muscle thick filament

(Nongthomba et al., 2004)

indirect flight muscle & striated muscle thin filament

(Nongthomba et al., 2004)

indirect flight muscle & Z disc

(Nongthomba et al., 2004)

indirect flight muscle cell

(Nikonova et al., 2022, Singh et al., 2014, Nongthomba et al., 2004, Nongthomba et al., 2003, Naimi et al., 2001, Barthmaier and Fyrberg, 1995, Prado et al., 1995)

indirect flight muscle cell, with Scer\GAL4^Hk-UH3, wupA^UAS.L9

(Singh et al., 2014)

indirect flight muscle cell (with wupA^fliH)

(Firdaus et al., 2015)

lateral oblique dorsal muscle cell

(Barbas et al., 1993)

mesothoracic tergotrochanter muscle cell

(Singh et al., 2014, Nongthomba et al., 2004, de la Pompa, 1994, Barbas et al., 1993)

muscle cell of coxal tergal remotor muscle 48a

(de la Pompa, 1994)

muscle cell of coxal tergal remotor muscle 48b

(de la Pompa, 1994)

muscle cell of dorsal longitudinal indirect flight muscle 45a

(de la Pompa, 1994)

muscle cell of dorsal longitudinal indirect flight muscle 45b

(de la Pompa, 1994)

muscle cell of dorsal longitudinal indirect flight muscle 45c

(de la Pompa, 1994)

muscle cell of dorsal longitudinal indirect flight muscle 45d

(de la Pompa, 1994)

muscle cell of dorsal longitudinal indirect flight muscle 45e

(de la Pompa, 1994)

muscle cell of dorsal longitudinal indirect flight muscle 45f

(de la Pompa, 1994)

muscle cell of lateral oblique dorsal muscle 46a

(de la Pompa, 1994)

muscle cell of lateral oblique dorsal muscle 46b

(de la Pompa, 1994)

muscle cell of tergosternal muscle 47a

(de la Pompa, 1994)

muscle cell of tergosternal muscle 47b

(de la Pompa, 1994)

muscle cell of tergosternal muscle 47c

(de la Pompa, 1994)

myofibril

(Beall and Fyrberg, 1991)

striated muscle thin filament

(Beall and Fyrberg, 1991)

tergosternal muscle cell

(Barbas et al., 1993)

wing

(Casas-Tintó and Ferrús, 2021, Nongthomba et al., 2003, de la Pompa, 1994, Homyk and Emerson, 1988, Deak et al., 1982)

Z disc

(Beall and Fyrberg, 1991)

Detailed Description

Statement

Reference

wupA^hdp-3 mutants exhibit held-up wings.

(Casas-Tintó and Ferrús, 2021)

wupA^fliH fails to complement wupA^hdp-3 and all the flies show muscle tearing and hypercontraction.

(Firdaus et al., 2015)

During the development of indirect flight muscle fibres, wupA^hdp-3 mutant pupal myoblasts fuse with dorsal medial muscle templates as in wild-type pupa. Following fusion, myotube formation and fibre shortening takes place but elongation of fibres fails to occur, leaving wupA^hdp-3 myotubes shorter than in wild type. While wild-type muscle fibres extend completely to form the cuticle, wupA^hdp-3 mutant dorsal medial muscle fibres are pulled from their attachment sites and start to degrade; with time, complete degradation occurs resulting in newly eclosed adults with no fibres. At 40-42 hours after pupal formation (APF) wupA^hdp-3 mutant myofibrils are clumped as filamentous bundles with no clear sarcomere-like structures and with visible thick and thin filaments that are scattered without regular structure, in contrast to wild-type myofibrils, which consist of thick and thin filaments assembled into clearly demarcated repetitive I-A-I bands. At this time, wild-type myofibrils exhibit uniform length sarcomeres demarcated by straight, electron-dense Z-discs, while wupA^hdp-3 mutant myofibrils have no Z-discs but contain stress-fibre-like structures, the usual precursor of Z-discs. These are abnormally large and may contain Z-disc-like electron dense bodies. By 46-48 hours APF, wupA^hdp-3 mutants have no Z-discs or sarcomeres. Heterozygous wupA^hdp-3 mutant indirect flight muscle fibres show a hypercontraction muscle phenotype in which the muscles are pulled to one of the attachment sites starting from the middle of the fibres. No such contraction is seen in wild-type controls. The outer areas of the wupA^hdp-3 heterozygote myofibrils have loosely packed thick-thin filaments and contain Z-discs and M-lines that appear streamed compared to wild type. The thorax of wupA^hdp-3 homozygous mutant flies has no visible indirect flight muscle or tergal depressor of trochanter muscle.

(Nongthomba et al., 2004)

100% of wupA^hdp-3/Y adults have an upheld wing phenotype. The indirect flight muscles fail to form and only small muscle remnants are seen in the thoraces of later pupal and adult stages.

(Nongthomba et al., 2003)

Flight muscles show abnormalities not long after myoblast fusion, partially elongate and only small muscle remnants remain in later pupae.

(Barthmaier and Fyrberg, 1995)

IFM phenotype cannot be suppressed by wupA^D3.

(Prado et al., 1995)

Wings are held up vertically, flies do not jump and walk weakly. Fibrillar muscles (dorsal-longitudinal muscle, DLM and dorso-ventral muscle, DVM) are reduced and the tergal depressor of trochanter muscle (TDT) is defective. Heterozygotes with wupA⁸³⁸⁴ have only remnants of the degenerating fibrillar muscles and no (TDT).

(de la Pompa, 1994)

Extreme allele. The development of the DLM, DVM and TDT muscles is aborted. Newly eclosed adults show occasional remnants of muscle tissue without any trace of sarcomeric organization. All other somatic and visceral muscles appear normal in structure. wupA^hdp-3/+ show abnormal muscle structure and function, wupA^hdp-3/Y; Dp(1;3)JC153/+ are wild type but wupA^hdp-3/Y; Dp(1;3)JC153/Dp(1;3)JC153 flies are unable to jump or fly (and show reduced viability, though their muscle histology and wing position are normal).

(Barbas et al., 1993)

In flight muscles of recently eclosed adults myofibrils are missing. Thick and thin filaments are present in the sarcoplasm though normal Z discs are not evident.

(Beall and Fyrberg, 1991)

Heterozygotes are flightless with a normal wing posture. Homozygotes are flightless with an abnormal wing posture. Most double heterozygotes with up^int-3 and some with up¹⁰¹ have an abnormal wing posture.

(Homyk and Emerson, 1988)

Flies are flightless and hold their wings in an abnormal position, and sometimes show variable jump ability.

(Deak et al., 1982)

External Data

Linkouts

Interactions

Show genetic interaction network for Enhancers & Suppressors

Phenotypic Class

Suppressed by

Statement

Reference

wupA^hdp-3 has visible | adult stage phenotype, suppressible by Dp(1;2)CH322-143G12

(Casas-Tintó and Ferrús, 2021)

wupA^hdp-3 has flightless phenotype, suppressible by Dp(1;3)JC153

(Casas-Tintó and Ferrús, 2021)

wupA^hdp-3 has flightless phenotype, suppressible by Dp(1;2)CH322-143G12

(Casas-Tintó and Ferrús, 2021)

wupA^hdp-3 has visible | adult stage phenotype, suppressible by Dp(1;3)JC153

(Casas-Tintó and Ferrús, 2021)

wupA^hdp-3 has visible phenotype, suppressible by Mhc¹²/Mhc^D41

(Nongthomba et al., 2003)

wupA^hdp-3 has visible phenotype, suppressible by Mhc¹²/Mhc^2D

(Nongthomba et al., 2003)

wupA^hdp-3 has visible phenotype, suppressible by Mhc¹²/Mhc¹²

(Nongthomba et al., 2003)

NOT suppressed by

Statement

Reference

wupA^hdp-3 has lethal | recessive phenotype, non-suppressible by mir-969^{sponge.V2.UAS.mCherry}/Scer\GAL4^αTub84B.PL

(Casas-Tintó and Ferrús, 2021)

wupA^hdp-3 has visible phenotype, non-suppressible by Mhc¹²/Mhc^2B

(Nongthomba et al., 2003)

wupA^hdp-3 has visible phenotype, non-suppressible by Mhc¹²/Mhc^2F

(Nongthomba et al., 2003)

wupA^hdp-3 has visible phenotype, non-suppressible by Mhc^D1/Mhc¹²

(Nongthomba et al., 2003)

wupA^hdp-3 has visible phenotype, non-suppressible by Mhc¹²/Mhc^D41

(Nongthomba et al., 2003)

wupA^hdp-3 has visible phenotype, non-suppressible by Mhc¹²/Mhc^D62

(Nongthomba et al., 2003)

Other

Statement

Reference

l(1)16Fe¹, wupA^hdp-3 has visible | dominant phenotype

(Prado et al., 1999)

l(1)16Fe¹, wupA^hdp-3 has abnormal flight | dominant phenotype

(Prado et al., 1999)

Phenotype Manifest In

Suppressed by

Statement

Reference

wupA^hdp-3 has indirect flight muscle cell phenotype, suppressible by Mhc¹²/Mhc^2F

(Nongthomba et al., 2003)

wupA^hdp-3 has indirect flight muscle cell phenotype, suppressible by Mhc¹²/Mhc¹²

(Nongthomba et al., 2003)

wupA^hdp-3 has indirect flight muscle cell phenotype, suppressible by Mhc¹²/Mhc^D62

(Nongthomba et al., 2003)

wupA^hdp-3 has wing phenotype, suppressible by Mhc¹²/Mhc^D41

(Nongthomba et al., 2003)

wupA^hdp-3 has wing phenotype, suppressible by Mhc¹²/Mhc¹²

(Nongthomba et al., 2003)

wupA^hdp-3 has wing phenotype, suppressible by Mhc¹²/Mhc^2D

(Nongthomba et al., 2003)

NOT suppressed by

Statement

Reference

wupA^hdp-3 has wing phenotype, non-suppressible by Mhc¹²/Mhc^2B

(Nongthomba et al., 2003)

wupA^hdp-3 has wing phenotype, non-suppressible by Mhc¹²/Mhc^2F

(Nongthomba et al., 2003)

wupA^hdp-3 has wing phenotype, non-suppressible by Mhc^D1/Mhc¹²

(Nongthomba et al., 2003)

wupA^hdp-3 has wing phenotype, non-suppressible by Mhc¹²/Mhc^D41

(Nongthomba et al., 2003)

wupA^hdp-3 has wing phenotype, non-suppressible by Mhc¹²/Mhc^D62

(Nongthomba et al., 2003)

wupA^hdp-3 has indirect flight muscle cell phenotype, non-suppressible by Mhc¹²/Mhc^2D

(Nongthomba et al., 2003)

wupA^hdp-3 has indirect flight muscle cell phenotype, non-suppressible by Mhc¹²/Mhc^2F

(Nongthomba et al., 2003)

wupA^hdp-3 has indirect flight muscle cell phenotype, non-suppressible by Mhc^D1/Mhc¹²

(Nongthomba et al., 2003)

wupA^hdp-3 has indirect flight muscle cell phenotype, non-suppressible by Mhc¹²/Mhc^D41

(Nongthomba et al., 2003)

wupA^hdp-3 has indirect flight muscle cell phenotype, non-suppressible by Mhc¹²/Mhc^D45

(Nongthomba et al., 2003)

wupA^hdp-3 has indirect flight muscle cell phenotype, non-suppressible by Tm2^D53

(Naimi et al., 2001)

Suppressor of

Statement

Reference

wupA^hdp-3 is a suppressor | partially of indirect flight muscle cell phenotype of Rbfox1^RNAi.UAS, Scer\GAL4^Mef2.PR

(Nikonova et al., 2022)

wupA^hdp-3 is a suppressor | partially of muscle cell | decreased number phenotype of Rbfox1^RNAi.UAS, Scer\GAL4^Mef2.PR

(Nikonova et al., 2022)

wupA^hdp-3 is a suppressor | partially of dorsal longitudinal indirect flight muscle cell phenotype of Rbfox1^RNAi.UAS, Scer\GAL4^Mef2.PR

(Nikonova et al., 2022)

Other

Statement

Reference

l(1)16Fe¹, wupA^hdp-3 has wing phenotype

(Prado et al., 1999)

l(1)16Fe¹, wupA^hdp-3 has adult somatic muscle cell phenotype

(Prado et al., 1999)

l(1)16Fe²³⁴³⁷, l(1)16Fm²³⁴³⁷, wupA^hdp-3 has adult somatic muscle cell phenotype

(Prado et al., 1999)

Mhc⁷, wupA^hdp-3 has striated muscle thin filament phenotype

(Beall and Fyrberg, 1991)

Additional Comments

Genetic Interactions

Statement

Reference

Mhc¹⁰/Mhc¹⁰ flies expressing Mhc^{R57-24.Act88F} completely restore indirect flight muscle fibre defects to a normal structural condition in wupA^hdp-3/+ flies.

The thorax of wupA^hdp-3/Y; Mhc¹⁰/+ flies have a small bunch of visible muscle mass, in contrast to wupA^hdp-3 ; Mhc+/+ flies which have no visible indirect flight muscle. wupA^hdp-3/Y; Mhc¹⁰/+ flies that express one copy of Mhc^{R57-24.Act88F} show a greater increase in muscle mass. This increase is greater still when flies express two copies of Mhc^{R57-24.Act88F}. Only in wupA^hdp-3/Y; Mhc¹⁰/Mhc¹⁰ flies that express two copies of Mhc^{R57-24.Act88F} can actual myofibrillar structures be seen.

In the thorax of wupA^hdp-3/Y; Mhc⁷/Mhc^2B flies there is an absence of myofibrils and no clear areas of interdigitated thick-thin filaments. There are visible "tiger-tail" structures formed by condensed serially repeated Z-discs connected by short stretches of thin filaments.

In wupA^hdp-3/Y; Mhc¹⁰/Mhc¹⁰; Mhc^{R57-24.Act88F} flies thick filaments, some thin filaments, isolated Z-discs and tiger-tails are readily visible but sarcomeric structures fail to assemble.

(Nongthomba et al., 2004)

41% of wupA^hdp-3/Y ; Mhc¹²/Mhc¹² flies have an upheld wing phenotype and 59% hold their wings normally. 41% of wupA^hdp-3/Y ; Mhc¹²/Mhc^2D flies have an upheld wing phenotype and 59% hold their wings normally. 61% of wupA^hdp-3/Y ; Mhc¹²/Mhc^D45 flies have an upheld wing phenotype and 39% hold their wings normally. The indirect flight muscles of wupA^hdp-3/Y ; Mhc¹²/Mhc^2B and wupA^hdp-3/Y ; Mhc¹²/Mhc^2F flies show a partial hypercontraction phenotype.

(Nongthomba et al., 2003)

l(1)16Fe¹ fails to complement wupA^hdp-3 with respect to wing position, flight and jump behaviour and muscle structure. The '23437' chromosome (in which l(1)16Fm and l(1)16Fe are affected) fails to complement wupA^hdp-3, resulting in severe muscle defects.

(Prado et al., 1999)

Z disc morphology is improved in the double mutant with Mhc⁷. Thin filaments are reduced, even in the absence of myosin.

(Beall and Fyrberg, 1991)

Xenogenetic Interactions

Statement

Reference

Complementation and Rescue Data

Partially rescued by

wupA^hdp-3 is partially rescued by wupA^UASp.B/Scer\GAL4^αTub84B.PL

(Casas-Tintó and Ferrús, 2021)

wupA^hdp-3 is partially rescued by wupA^UAS.L9/Scer\GAL4^Hk-UH3

(Singh et al., 2014)

Not rescued by

wupA^hdp-3 is not rescued by wupA^UASp.A/Scer\GAL4^αTub84B.PL

(Casas-Tintó and Ferrús, 2021)

wupA^hdp-3 is not rescued by Scer\GAL4^αTub84B.PL/wupA^UASp.C

(Casas-Tintó and Ferrús, 2021)

wupA^hdp-3 is not rescued by wupA^UASp.D/Scer\GAL4^αTub84B.PL

(Casas-Tintó and Ferrús, 2021)

wupA^hdp-3 is not rescued by wupA^UASp.E/Scer\GAL4^αTub84B.PL

(Casas-Tintó and Ferrús, 2021)

wupA^hdp-3 is not rescued by wupA^UASp.F/Scer\GAL4^αTub84B.PL

(Casas-Tintó and Ferrús, 2021)

wupA^hdp-3 is not rescued by wupA^UASp.G/Scer\GAL4^αTub84B.PL

(Casas-Tintó and Ferrús, 2021)

wupA^hdp-3 is not rescued by Scer\GAL4^αTub84B.PL/wupA^UASp.H

(Casas-Tintó and Ferrús, 2021)

wupA^hdp-3 is not rescued by wupA^UASp.I/Scer\GAL4^αTub84B.PL

(Casas-Tintó and Ferrús, 2021)

wupA^hdp-3 is not rescued by wupA^UASp.J/Scer\GAL4^αTub84B.PL

(Casas-Tintó and Ferrús, 2021)

wupA^hdp-3 is not rescued by wupA^UASp.K/Scer\GAL4^αTub84B.PL

(Casas-Tintó and Ferrús, 2021)

wupA^hdp-3 is not rescued by wupA^{fTRG00925.sfGFP-TVPTBF}

(Casas-Tintó and Ferrús, 2021)

Comments

wupA^fliH fails to complement wupA^hdp-3 and all the flies show muscle tearing and hypercontraction.

(Firdaus et al., 2015)

Expression of wupA^Scer\UAS.L9 under the control of Scer\GAL4^Hk-UH3 rescues the structural defects seen in the indirect flight muscles of hemizygous wupA^hdp-3 mutants, though not completely. Rescued hemizygous flies are completely flightless and the size of the myofibrils is comparatively smaller compared to controls. Rescued heterozygous flies show a better myofibrillar structure where the sarcomeres look near-normal, but are still unable to fly.

(Singh et al., 2014)

Images (0)

Mutant

Wild-type

Stocks (0)

Notes on Origin

Discoverer

Comments

Mutant muscle phenotype is due to lack of one or more TnI isoforms.

(de la Pompa, 1994)

External Crossreferences and Linkouts ( 0 )

Synonyms and Secondary IDs (4)

Reported As

Symbol Synonym

hdp³

(Casas-Tintó and Ferrús, 2021, Singh et al., 2014, Nongthomba et al., 2004, Nongthomba et al., 2003, Nongthomba and Sparrow, 2002, Naimi et al., 2001, Kronert et al., 1999, Barthmaier and Fyrberg, 1995, Prado et al., 1995, de la Pompa, 1994, Barbas et al., 1991, Homyk and Emerson, 1988, Deak et al., 1982)

wupA^hdp-3

(Chatterjee et al., 2016, Firdaus et al., 2015)

wupA^hdp3

(Nikonova et al., 2022, Prado et al., 1999)

Name Synonyms

heldup3

(Barbas et al., 1993, Beall and Fyrberg, 1991)

Secondary FlyBase IDs

References (20)

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