FlyBase Allele Report: Dmel\wupA[hdp-2]

General Information

Symbol

Dmel\wupA^hdp-2

Species

D. melanogaster

Name

FlyBase ID

FBal0033188

Feature type

allele

Associated gene

Dmel\wupA

Associated Insertion(s)

Carried in Construct

Also Known As

hdp², hdp2, heldup², wupA^hdp2

Key Links

Genomic Maps

JBrowse

Allele class

Mutagen

Nature of the Allele

Allele class

Mutagen

Progenitor genotype

Cytology

Description

Amino acid replacement: A116V. A116V change falls in the constitutive exon 5.

(Prado et al., 1995)

Nucleotide substitution: C?T. Amino acid replacement: A55V. Codon 55 is an invariant residue in troponin-I isoforms, including those of vertebrates.

(Beall and Fyrberg, 1991)

Mutations Mapped to the Genome

Curation Data

Type

Location

Additional Notes

References

point_mutation

X:18,113,362..18,113,362 [-]

Nucleotide change:

C18113362T

Reported nucleotide change:

C?T

Amino acid change:

Reported amino acid change:

A55V, A116V

(Prado et al., 1995, Beall and Fyrberg, 1991)

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 cardiomyopathy

CEA

(Gautam et al., 2015, Naimi et al., 2001)

model of dilated cardiomyopathy

CEA

(Abraham and Wolf, 2013, Lin et al., 2011, Wolf et al., 2006)

model of heart disease

CEA

(Choma et al., 2011)

Modifiers Based on Experimental Evidence ( 1 )

Disease

Interaction

References

model of dilated cardiomyopathy

is ameliorated by SERCA^wt.tinC

(Abraham and Wolf, 2013)

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

Disease-implicated variant(s)

Phenotypic Data

Phenotypic Class

Phenotype Manifest In

adult heart

(Lee et al., 2014, Abraham and Wolf, 2013, Lin et al., 2011, Wolf et al., 2006)

adult heart chamber

(Wolf et al., 2006)

adult heart muscle cell

(Wolf et al., 2006)

basement membrane | ectopic

(Wolf et al., 2006)

coxal tergal remotor muscle cell

(Deak et al., 1982)

dorsal longitudinal indirect flight muscle cell

(Kronert et al., 1999, Deak et al., 1982)

dorsal medial muscle & sarcomere

(Kronert et al., 1999)

dorsal medial muscle & striated muscle thick filament

(Kronert et al., 1999)

dorsal medial muscle & striated muscle thin filament

(Kronert et al., 1999)

embryonic/larval heart

(Choma et al., 2011)

embryonic somatic muscle & Z disc (with wupA^D3)

(Marin et al., 2004)

indirect flight muscle & striated muscle thick filament

(Naimi et al., 2001)

indirect flight muscle & striated muscle thin filament

(Naimi et al., 2001)

indirect flight muscle & Z disc (with wupA^D3)

(Marin et al., 2004)

indirect flight muscle cell

(Firdaus et al., 2015, Gautam et al., 2015, Wolf et al., 2006, Nongthomba et al., 2003, Naimi et al., 2001, Prado et al., 1995)

lateral oblique dorsal muscle cell

(Deak et al., 1982)

mesothoracic leg

(Deak, 1977)

mesothoracic tergotrochanter muscle cell

(Elliott et al., 2007, Prado et al., 1995, Barbas et al., 1991, Deak et al., 1982, Deak, 1977)

muscle cell of coxal tergal remotor muscle 48a

(Barbas et al., 1991, Deak, 1977)

muscle cell of coxal tergal remotor muscle 48b

(Barbas et al., 1991, Deak, 1977)

muscle cell of dorsal longitudinal indirect flight muscle 45a

(Kronert et al., 1999, Barbas et al., 1991)

muscle cell of dorsal longitudinal indirect flight muscle 45b

(Kronert et al., 1999, Barbas et al., 1991)

muscle cell of dorsal longitudinal indirect flight muscle 45c

(Kronert et al., 1999, Barbas et al., 1991)

muscle cell of dorsal longitudinal indirect flight muscle 45d

(Kronert et al., 1999, Barbas et al., 1991)

muscle cell of dorsal longitudinal indirect flight muscle 45e

(Kronert et al., 1999, Barbas et al., 1991)

muscle cell of dorsal longitudinal indirect flight muscle 45f

(Kronert et al., 1999, Barbas et al., 1991)

muscle cell of lateral oblique dorsal muscle 46a

(Barbas et al., 1991, Deak, 1977)

muscle cell of lateral oblique dorsal muscle 46b

(Barbas et al., 1991, Deak, 1977)

muscle cell of leg muscle of leg

(Naimi et al., 2001)

muscle cell of tergosternal muscle 47a

(Barbas et al., 1991, Deak, 1977)

muscle cell of tergosternal muscle 47b

(Barbas et al., 1991, Deak, 1977)

muscle cell of tergosternal muscle 47c

(Barbas et al., 1991, Deak, 1977)

myofibril

(Deak et al., 1982)

sarcomere

(Beall and Fyrberg, 1991, Deak et al., 1982)

tergosternal muscle cell

(Deak et al., 1982)

wing

(Casas-Tintó and Ferrús, 2021, Nongthomba et al., 2003, Naimi et al., 2001, Homyk and Emerson, 1988, Deak, 1977)

Z disc

(Beall and Fyrberg, 1991, Deak et al., 1982)

Detailed Description

Statement

Reference

wupA^hdp-2 mutants exhibit held-up wings.

(Casas-Tintó and Ferrús, 2021)

Homozygous wupA^hdp-2 mutant flies show indirect flight muscle hypercontraction.

(Firdaus et al., 2015)

Raman spectroscopy reveals that, as flies age, wupA^hdp-2 mutant muscles show signs of denaturation and overall decay in protein levels, which is not observed in wild-type controls.

(Gautam et al., 2015)

wupA^hdp-2/+ mutant adults exhibit cardiac dilation and reduced fractional shortening, as compared to controls.

(Lee et al., 2014)

wupA^hdp-2 mutant flies have significantly reduced cardiac function compared to controls. Heart rate is also mildly reduced.

(Abraham and Wolf, 2013)

wupA^hdp-2 hearts have normal fractional shortening but diminished systolic and diastolic wall velocities compared with wild-type pre-pupal hearts.

(Choma et al., 2011)

wupA^hdp-2 mutant hearst exhibit abnormalities in myocardial Ca[2+] handling that include an increase in the duration of the 50% rise in intensity to peak intensity, the half-time of fluorescence decline from peak, the full duration at half-maximal intensity, and decreases in the linear slope of decay from 80% to 20% intensity decay.

Hearts from wupA^hdp-2 mutants exhibit a reduction in caffeine-induced Ca[2+] elevation.

(Lin et al., 2011)

The jump muscle output of 4 day old wupA^hdp-2 flies (measured using displacement of an ergometer) is decreased compared to control flies.

(Elliott et al., 2007)

wupA^hdp-2 mutant heterozygotes demonstrate impaired systolic function with preserved distolic dimensions, whereas wupA^hdp-2 mutant homozygotes exhibit both impaired systolic function and cardiac enlargement (recapitulating the clinical phenotype of human dilated cardiomyopathy). The outer and inner muscle layers of the cardiac chamber of homozygote wupA^hdp-2 mutants are markedly abnormal. The outer muscle layer is considerably thinner than in controls with redundant basal lamina in the space between the outer and inner walls. The mutant myocardium shows considerable disorientation of myofibrils throughout the inner layer and displays swollen sarcoplasmic reticulum when compared to controls. However, the T-tubule dyads appear unaffected in the mutant, and the ratio of thin and thick filaments in the myocardium of homozygous wupA^hdp-2 mutants is similar to controls. The myofibrils of the indirect flight muscles exhibit marked deterioration in wupA^hdp-2 homozygotes compared to controls.

(Wolf et al., 2006)

wupA^hdp-2/wupA^D3 embryos have only subtle abnormalities in the somatic muscles, such as disordered alignment of Z discs and failure to anchor all thin filaments from a sarcomere. The indirect flight muscles of wupA^hdp-2/wupA^D3 adults show abnormalities in the central part of some Z discs.

(Marin et al., 2004)

The thick and thin filaments of the indirect flight muscles of hemizygous animals are normal before 78 hours after puparium formation (APF), but are hypercontracted after 78 hours APF. The fibres are bunched at one end of the thorax in 2-4 day old adults. 100% of hemizygous male adults have an upheld wing phenotype and show hypercontraction of the indirect flight muscle filaments. wupA^hdp-2 flies show age-related myopathy in their walking behaviour.

(Nongthomba et al., 2003)

Indirect flight muscles (IFMs) progressively hypercontract in mutant flies (hypercontraction begins 78 hours after puparium formation), so that myofibrillar material remains only near the muscle attachment sites in newly emerged flies. 2-3 day old mutant flies show an almost complete absence of wild-type myofibrillar structure in the IFMs. The fibres contain disorganised thick and thin filaments with only small islands of myofibrillar lattice. Homozygous and hemizygous flies hold their wings vertically. The walk speed of wupA^hdp-2 flies is reduced in young flies compared to wild type and reduces further with age. Older flies appear crippled, continually fall over or show a propensity to fall off vertical surfaces. Most leg muscles of 12 day old mutant flies lack myofilaments and have large intracellular vacuoles and electron dense staining of cells and nuclei. Detachment of muscles from apodemes is seen. Crawling behaviour of wupA^hdp-2 larvae is very different from wild type. Many appear paralysed in their posterior segments; often the posterior is raised. The larvae "roll" as they crawl. The number of larval feeding movements/minute is reduced compared to wild type.

(Naimi et al., 2001)

The six dorsolongitudinal muscles (DLMs) appear torn apart from the centre in wupA^hdp-2 mutant flies. In the remaining muscle material near the attachment sites, the length of the sarcomere is reduced by 40% and the thick-thin filament pattern is destroyed (mainly due to the collapse of the thin filaments). Hemizygous males have heldout wings.

(Kronert et al., 1999)

Indirect flight muscles (IFM) show near normal development during metamorphosis. During the first day posteclosion, the muscles collapse and only hypercontracted stumps are left near the attachment points to the cuticle. As a result, the flies hold their wings in an up position with full penetrance and expressivity. Tubular muscles in the body wall have minor or unnoticeable defects and still allow proper function. The TDT shows an intermediate degree of alteration that most likely explains the absence of the jump response. Sarcomere length is about half of wild type, the M band is not detected. Males and females of wupA^hdp-2/wupA^D3 genotype show a normal wing position in 95% cases, the remaining 5% still show a variety of abnormal wing positions. The IFM and TDT muscles are restored to a great extent. Sarcomere structure is also restored thought they are about 25% longer than wild type. Flight performance and jump ability demonstrate a functional recovery of the muscles.

(Prado et al., 1995)

Alters the dorso-longitudinal muscles (DLM) and dorso-ventral muscles (DVM) and slightly alters the tergal depressor of the trochanter muscle (TDT). This phenotype becomes more extreme when transheterozygous with l(1)16Fe¹.

(Barbas et al., 1991)

In flight muscles of recently eclosed adults myofibrils are recognizable, but disruptions of sarcomeric organization are readily apparent. Myofibrils form normally until they are approximately two-thirds of their final diameter. Viewed longitudinally, a slight perturbation of Z-disc morphology is evident. Viewed in cross section, myofilament lattices are fairly normal, though inconsistencies in packing arrangement are frequent. During the final day of adult muscle development the myofibrils progressively degenerate. By the time the adult ecloses thick and thin filaments have moved out of lateral register, causing skewing of M-lines and Z-discs.

(Beall and Fyrberg, 1991)

Normal electroretinogram.

(Homyk and Pye, 1989)

Homozygotes are flightless, and have an abnormal wing posture.

(Homyk and Emerson, 1988)

Muscle degeneration mutant. DVMs, DLMs and TDT are reduced. Myofibrillar structure is disrupted and Z bands are highly disturbed.

(Deak et al., 1982)

wupA^hdp-2 mutants show 60% viability.

wupA^hdp-2 flies hold their wings up vertically but are otherwise morphologically normal, except for bent femurs in 5% of mutants. Mutants are flightless, jumpless and are only able to walk weakly. Excitation results in folding of the mesothoracic legs and walking on only the pro- and metathoracic legs. When wupA^hdp-2 flies are etherized, the mesothoracic legs remain extended, while all three pairs of legs fold up in wild-type flies.

wupA^hdp-2 flies are lacking many indirect flight muscles. Only the insertions of the dorsal longitudinal fibres are present and occasionally fibres 5 and 6 are present, although these are very abnormal. In all flies, dorsoventral fibres (DVMs) 7, 8 and 9 are absent, while some of the posterior DVMs are present in many wupA^hdp-2 mutants. All of the remaining muscle fibres lack glycogen and contain grossly distorted sarcomeres, thick and thin filaments and Z-bands.

During the first 60 hours after pupariation the muscles of wupA^hdp-2 flies appear to develop normally but at 60 hours the indirect flight muscle fibres extend to approximately three-quarters of the length of the thorax and then degenerate rapidly so that by emergence little of the flight musculature remains.

The tergal depressor of the trochanters (TDTs) are very abnormal in the majority of wupA^hdp-2 flies, although 1-2% of these flies have TDTs of wild-type appearance. In some wupA^hdp-2 flies, the TDTs are absent, while in others the fibres are detached from each other and the normally hollow core is filled with material.

(Deak, 1977)

External Data

Linkouts

Interactions

Show genetic interaction network for Enhancers & Suppressors

Phenotypic Class

Suppressed by

Statement

Reference

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

(Casas-Tintó and Ferrús, 2021)

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

(Casas-Tintó and Ferrús, 2021)

wupA^hdp-2 has visible | adult stage phenotype, suppressible by Dp(1;3)JC153

(Casas-Tintó and Ferrús, 2021)

wupA^hdp-2 has flightless phenotype, suppressible by Dp(1;3)JC153

(Casas-Tintó and Ferrús, 2021)

wupA^hdp-2 has visible phenotype, suppressible | partially by Mhc^{R57-24.Act88F}/Mhc¹⁰

(Nongthomba et al., 2003)

wupA^hdp-2 has abnormal locomotor behavior phenotype, suppressible by Mhc[+]/Mhc^2B

(Nongthomba et al., 2003)

wupA^hdp-2 has abnormal locomotor behavior phenotype, suppressible by Mhc^2D/Mhc^2D

(Nongthomba et al., 2003)

wupA^hdp-2 has abnormal locomotor behavior phenotype, suppressible by Mhc[+]/Mhc^2F

(Nongthomba et al., 2003)

wupA^hdp-2 has abnormal locomotor behavior phenotype, suppressible by Mhc[+]/Mhc^2D

(Nongthomba et al., 2003)

wupA^hdp-2 has visible phenotype, suppressible by Mhc[+]/Mhc^2B

(Nongthomba et al., 2003)

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

(Nongthomba et al., 2003)

wupA^hdp-2 has visible phenotype, suppressible by Mhc[+]/Mhc^2D

(Nongthomba et al., 2003)

wupA^hdp-2 has visible phenotype, suppressible by Mhc[+]/Mhc^2F

(Nongthomba et al., 2003)

wupA^hdp-2 has visible phenotype, suppressible by Mhc^D1/Mhc[+]

(Nongthomba et al., 2003)

wupA^hdp-2 has visible phenotype, suppressible by Mhc[+]/Mhc^D41

(Nongthomba et al., 2003)

wupA^hdp-2 has visible phenotype, suppressible by Mhc[+]/Mhc^D45

(Nongthomba et al., 2003)

wupA^hdp-2 has visible phenotype, suppressible by Mhc[+]/Mhc^D62

(Nongthomba et al., 2003)

wupA^hdp-2 has visible phenotype, suppressible by Mhc[+]/Mhc^{R57-24.Act88F}

(Nongthomba et al., 2003)

wupA^hdp-2 has visible phenotype, suppressible by Su(2)E[+]/Su(2)E^E

(Nongthomba et al., 2003)

wupA^hdp-2 has visible phenotype, suppressible by Mhc[+]/Mhc¹⁰

(Nongthomba et al., 2003)

wupA^hdp-2 has abnormal jumping phenotype, suppressible by Tm2^D53/Tm2[+]

(Naimi et al., 2001)

wupA^hdp-2 has abnormal jumping phenotype, suppressible by Tm2^D53/Tm2^D53

(Naimi et al., 2001)

wupA^hdp-2 has abnormal locomotor behavior | larval stage phenotype, suppressible by Tm2^D53

(Naimi et al., 2001)

wupA^hdp-2 has abnormal locomotor behavior phenotype, suppressible by Tm2^D53/Tm2[+]

(Naimi et al., 2001)

wupA^hdp-2 has visible phenotype, suppressible by Tm2[+]/Tm2^DL2

(Naimi et al., 2001)

wupA^hdp-2 has visible phenotype, suppressible by Tm2^C10/Tm2[+]

(Naimi et al., 2001)

wupA^hdp-2 has visible | recessive phenotype, suppressible by Mhc^D1

(Kronert et al., 1999)

wupA^hdp-2 has visible | recessive phenotype, suppressible by Mhc^D41

(Kronert et al., 1999)

wupA^hdp-2 has visible | recessive phenotype, suppressible by Mhc^D45

(Kronert et al., 1999)

wupA^hdp-2 has visible | recessive phenotype, suppressible by Mhc^D62

(Kronert et al., 1999)

NOT suppressed by

Statement

Reference

wupA^hdp-2 has abnormal size | dominant | adult stage phenotype, non-suppressible by Galk^MB10638/Galk[+]

(Lee et al., 2014)

wupA^hdp-2 has flightless phenotype, non-suppressible by Mhc[+]/Mhc¹⁰

(Nongthomba et al., 2003)

wupA^hdp-2 has flightless phenotype, non-suppressible by Mhc[+]/Mhc^{R57-24.Act88F}

(Nongthomba et al., 2003)

wupA^hdp-2 has flightless phenotype, non-suppressible by Mhc^{R57-24.Act88F}/Mhc¹⁰

(Nongthomba et al., 2003)

wupA^hdp-2 has visible phenotype, non-suppressible by +/Df(3R)ea-5022rx1

(Naimi et al., 2001)

wupA^hdp-2 has visible phenotype, non-suppressible by Tm2[+]/Tm2^J8

(Naimi et al., 2001)

wupA^hdp-2 has visible phenotype, non-suppressible by Tm2^L2/Tm2[+]

(Naimi et al., 2001)

Suppressor of

Statement

Reference

wupA^hdp-2 is a suppressor of flightless phenotype of Act88F⁶, Act88F^D292V

(Sevdali et al., 2013)

Other

Statement

Reference

Mhc^S1, wupA^hdp-2 has lethal phenotype

(Montana and Littleton, 2004)

Mhc^S2, wupA^hdp-2 has lethal phenotype

(Montana and Littleton, 2004)

Mhc^D41, wupA^hdp-2 has abnormal flight phenotype

(Nongthomba et al., 2003)

Mhc^D45, wupA^hdp-2 has abnormal flight phenotype

(Nongthomba et al., 2003)

Mhc^D62, wupA^hdp-2 has abnormal flight phenotype

(Nongthomba et al., 2003)

up¹⁰¹, wupA^hdp-2 has visible phenotype

(Nongthomba et al., 2003)

up¹⁰¹, wupA^hdp-2 has flightless phenotype

(Nongthomba et al., 2003)

Mhc^D1/Mhc[+], wupA^hdp-2 has abnormal flight phenotype

(Nongthomba et al., 2003)

Mhc[+]/Mhc^D41, wupA^hdp-2 has abnormal flight phenotype

(Nongthomba et al., 2003)

Mhc[+]/Mhc^D45, wupA^hdp-2 has abnormal flight phenotype

(Nongthomba et al., 2003)

Mhc[+]/Mhc^D62, wupA^hdp-2 has abnormal flight phenotype

(Nongthomba et al., 2003)

Mhc^D1, wupA^hdp-2 has abnormal flight phenotype

(Nongthomba et al., 2003)

Tm2^D53, wupA^hdp-2 has abnormal flight phenotype

(Naimi et al., 2001)

Tm2^D53/Tm2[+], wupA^hdp-2 has abnormal flight phenotype

(Naimi et al., 2001)

Mhc⁵, wupA^hdp-2 has lethal phenotype

(Kronert et al., 1999)

l(1)16Fe¹, wupA^hdp-2 has visible | dominant phenotype

(Prado et al., 1999)

l(1)16Fe¹, wupA^hdp-2 has abnormal flight | dominant phenotype

(Prado et al., 1999)

Actn¹⁴, wupA^hdp-2 has flightless | dominant phenotype

(Homyk and Emerson, 1988)

Actn³, wupA^hdp-2 has flightless | dominant phenotype

(Homyk and Emerson, 1988)

Actn⁴, wupA^hdp-2 has flightless | dominant phenotype

(Homyk and Emerson, 1988)

up¹⁰¹, wupA^hdp-2 has visible | dominant phenotype

(Homyk and Emerson, 1988)

up¹⁰¹, wupA^hdp-2 has flightless | dominant phenotype

(Homyk and Emerson, 1988)

up^int-3, wupA^hdp-2 has visible | dominant phenotype

(Homyk and Emerson, 1988)

up^int-3, wupA^hdp-2 has flightless | dominant phenotype

(Homyk and Emerson, 1988)

up^x, wupA^hdp-2 has visible | dominant phenotype

(Homyk and Emerson, 1988)

up^x, wupA^hdp-2 has flightless | dominant phenotype

(Homyk and Emerson, 1988)

Actn⁸, wupA^hdp-2 has flightless | dominant phenotype

(Homyk and Emerson, 1988)

Mhc⁵, wupA^hdp-2 has visible | dominant phenotype

(Homyk and Emerson, 1988)

Mhc⁵, wupA^hdp-2 has flightless | dominant phenotype

(Homyk and Emerson, 1988)

Mhc⁸, wupA^hdp-2 has lethal | dominant phenotype

(Homyk and Emerson, 1988)

Phenotype Manifest In

Suppressed by

Statement

Reference

wupA^hdp-2 has adult heart phenotype, suppressible by SERCA^wt.tinC

(Abraham and Wolf, 2013)

wupA^hdp-2 has indirect flight muscle cell phenotype, suppressible by Mhc[+]/Mhc^D41

(Marin et al., 2004, Nongthomba et al., 2003)

wupA^hdp-2 has indirect flight muscle cell phenotype, suppressible | partially by Mhc^{R57-24.Act88F}/Mhc¹⁰

(Nongthomba et al., 2003)

wupA^hdp-2 has wing phenotype, suppressible | partially by Mhc^{R57-24.Act88F}/Mhc¹⁰

(Nongthomba et al., 2003)

wupA^hdp-2 has indirect flight muscle cell phenotype, suppressible by Mhc[+]/Mhc^2B

(Nongthomba et al., 2003)

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

(Nongthomba et al., 2003)

wupA^hdp-2 has indirect flight muscle cell phenotype, suppressible by Mhc^D1/Mhc[+]

(Nongthomba et al., 2003)

wupA^hdp-2 has indirect flight muscle cell phenotype, suppressible by Mhc[+]/Mhc^D45

(Nongthomba et al., 2003)

wupA^hdp-2 has indirect flight muscle cell phenotype, suppressible by Mhc[+]/Mhc^D62

(Nongthomba et al., 2003)

wupA^hdp-2 has indirect flight muscle cell phenotype, suppressible by Mhc[+]/Mhc¹⁰

(Nongthomba et al., 2003)

wupA^hdp-2 has indirect flight muscle cell phenotype, suppressible by Mhc[+]/Mhc^2D

(Nongthomba et al., 2003)

wupA^hdp-2 has indirect flight muscle cell phenotype, suppressible by Mhc[+]/Mhc^2F

(Nongthomba et al., 2003)

wupA^hdp-2 has indirect flight muscle cell phenotype, suppressible by Mhc[+]/Mhc^{R57-24.Act88F}

(Nongthomba et al., 2003)

wupA^hdp-2 has indirect flight muscle cell phenotype, suppressible by Su(2)E[+]/Su(2)E^E

(Nongthomba et al., 2003)

wupA^hdp-2 has wing phenotype, suppressible by Mhc[+]/Mhc^2B

(Nongthomba et al., 2003)

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

(Nongthomba et al., 2003)

wupA^hdp-2 has wing phenotype, suppressible by Mhc[+]/Mhc¹⁰

(Nongthomba et al., 2003)

wupA^hdp-2 has wing phenotype, suppressible by Mhc[+]/Mhc^2D

(Nongthomba et al., 2003)

wupA^hdp-2 has wing phenotype, suppressible by Mhc[+]/Mhc^2F

(Nongthomba et al., 2003)

wupA^hdp-2 has wing phenotype, suppressible by Mhc^D1/Mhc[+]

(Nongthomba et al., 2003)

wupA^hdp-2 has wing phenotype, suppressible by Mhc[+]/Mhc^D41

(Nongthomba et al., 2003)

wupA^hdp-2 has wing phenotype, suppressible by Mhc[+]/Mhc^D45

(Nongthomba et al., 2003)

wupA^hdp-2 has wing phenotype, suppressible by Mhc[+]/Mhc^D62

(Nongthomba et al., 2003)

wupA^hdp-2 has wing phenotype, suppressible by Mhc[+]/Mhc^{R57-24.Act88F}

(Nongthomba et al., 2003)

wupA^hdp-2 has wing phenotype, suppressible by Su(2)E[+]/Su(2)E^E

(Nongthomba et al., 2003)

wupA^hdp-2 has indirect flight muscle cell phenotype, suppressible by Tm2^D53/Tm2^D53

(Naimi et al., 2001)

wupA^hdp-2 has indirect flight muscle cell phenotype, suppressible by +/Df(3R)ea-5022rx1

(Naimi et al., 2001)

wupA^hdp-2 has indirect flight muscle cell phenotype, suppressible by Tm2^D53/Tm2[+]

(Naimi et al., 2001)

wupA^hdp-2 has indirect flight muscle cell phenotype, suppressible by Tm2[+]/Tm2^DL2

(Naimi et al., 2001)

wupA^hdp-2 has indirect flight muscle cell phenotype, suppressible by Tm2^C10/Tm2[+]

(Naimi et al., 2001)

wupA^hdp-2 has indirect flight muscle cell phenotype, suppressible by Tm2[+]/Tm2^J8

(Naimi et al., 2001)

wupA^hdp-2 has muscle cell of leg muscle of leg phenotype, suppressible by Tm2^D53/Tm2[+]

(Naimi et al., 2001)

wupA^hdp-2 has wing phenotype, suppressible by Tm2[+]/Tm2^DL2

(Naimi et al., 2001)

wupA^hdp-2 has wing phenotype, suppressible by Tm2^C10/Tm2[+]

(Naimi et al., 2001)

wupA^hdp-2 has muscle cell of dorsal longitudinal indirect flight muscle 45a phenotype, suppressible by Mhc^D1

(Kronert et al., 1999)

wupA^hdp-2 has muscle cell of dorsal longitudinal indirect flight muscle 45b phenotype, suppressible by Mhc^D1

(Kronert et al., 1999)

wupA^hdp-2 has muscle cell of dorsal longitudinal indirect flight muscle 45c phenotype, suppressible by Mhc^D1

(Kronert et al., 1999)

wupA^hdp-2 has muscle cell of dorsal longitudinal indirect flight muscle 45d phenotype, suppressible by Mhc^D1

(Kronert et al., 1999)

wupA^hdp-2 has muscle cell of dorsal longitudinal indirect flight muscle 45e phenotype, suppressible by Mhc^D1

(Kronert et al., 1999)

wupA^hdp-2 has muscle cell of dorsal longitudinal indirect flight muscle 45f phenotype, suppressible by Mhc^D1

(Kronert et al., 1999)

wupA^hdp-2 has muscle cell of dorsal longitudinal indirect flight muscle 45a phenotype, suppressible by Mhc^D41

(Kronert et al., 1999)

wupA^hdp-2 has muscle cell of dorsal longitudinal indirect flight muscle 45b phenotype, suppressible by Mhc^D41

(Kronert et al., 1999)

wupA^hdp-2 has muscle cell of dorsal longitudinal indirect flight muscle 45c phenotype, suppressible by Mhc^D41

(Kronert et al., 1999)

wupA^hdp-2 has muscle cell of dorsal longitudinal indirect flight muscle 45d phenotype, suppressible by Mhc^D41

(Kronert et al., 1999)

wupA^hdp-2 has muscle cell of dorsal longitudinal indirect flight muscle 45e phenotype, suppressible by Mhc^D41

(Kronert et al., 1999)

wupA^hdp-2 has muscle cell of dorsal longitudinal indirect flight muscle 45f phenotype, suppressible by Mhc^D41

(Kronert et al., 1999)

wupA^hdp-2 has muscle cell of dorsal longitudinal indirect flight muscle 45a phenotype, suppressible by Mhc^D45

(Kronert et al., 1999)

wupA^hdp-2 has muscle cell of dorsal longitudinal indirect flight muscle 45b phenotype, suppressible by Mhc^D45

(Kronert et al., 1999)

wupA^hdp-2 has muscle cell of dorsal longitudinal indirect flight muscle 45c phenotype, suppressible by Mhc^D45

(Kronert et al., 1999)

wupA^hdp-2 has muscle cell of dorsal longitudinal indirect flight muscle 45d phenotype, suppressible by Mhc^D45

(Kronert et al., 1999)

wupA^hdp-2 has muscle cell of dorsal longitudinal indirect flight muscle 45e phenotype, suppressible by Mhc^D45

(Kronert et al., 1999)

wupA^hdp-2 has muscle cell of dorsal longitudinal indirect flight muscle 45f phenotype, suppressible by Mhc^D45

(Kronert et al., 1999)

wupA^hdp-2 has muscle cell of dorsal longitudinal indirect flight muscle 45a phenotype, suppressible by Mhc^D62

(Kronert et al., 1999)

wupA^hdp-2 has muscle cell of dorsal longitudinal indirect flight muscle 45b phenotype, suppressible by Mhc^D62

(Kronert et al., 1999)

wupA^hdp-2 has muscle cell of dorsal longitudinal indirect flight muscle 45c phenotype, suppressible by Mhc^D62

(Kronert et al., 1999)

wupA^hdp-2 has muscle cell of dorsal longitudinal indirect flight muscle 45d phenotype, suppressible by Mhc^D62

(Kronert et al., 1999)

wupA^hdp-2 has muscle cell of dorsal longitudinal indirect flight muscle 45e phenotype, suppressible by Mhc^D62

(Kronert et al., 1999)

wupA^hdp-2 has muscle cell of dorsal longitudinal indirect flight muscle 45f phenotype, suppressible by Mhc^D62

(Kronert et al., 1999)

wupA^hdp-2 has phenotype, suppressible by Tm2^D53

(Sparrow et al., 1997)

NOT suppressed by

Statement

Reference

wupA^hdp-2 has adult heart phenotype, non-suppressible by Galk^MB10638/Galk[+]

(Lee et al., 2014)

wupA^hdp-2 has wing phenotype, non-suppressible by Tm2[+]/Tm2^J8

(Naimi et al., 2001)

wupA^hdp-2 has wing phenotype, non-suppressible by Tm2^L2/Tm2[+]

(Naimi et al., 2001)

wupA^hdp-2 has indirect flight muscle cell phenotype, non-suppressible by Tm2^L2/Tm2[+]

(Naimi et al., 2001)

wupA^hdp-2 has wing phenotype, non-suppressible by +/Df(3R)ea-5022rx1

(Naimi et al., 2001)

wupA^hdp-2 has phenotype, non-suppressible by Df(2L)H20

(Kronert et al., 1999)

wupA^hdp-2 has phenotype, non-suppressible by Mhc⁶

(Kronert et al., 1999)

wupA^hdp-2 has phenotype, non-suppressible by Mhc⁸

(Kronert et al., 1999)

Other

Statement

Reference

up¹⁰¹, wupA[+]/wupA^hdp-2 has indirect flight muscle cell phenotype

(Firdaus et al., 2015)

Mhc^S1, wupA[+]/wupA^hdp-2 has mesothoracic femur phenotype

(Montana and Littleton, 2004)

Mhc^S2, wupA[+]/wupA^hdp-2 has mesothoracic femur phenotype

(Montana and Littleton, 2004)

Mhc^S2, wupA^hdp-2 has mesothoracic femur phenotype

(Montana and Littleton, 2004)

Mhc^S1, wupA^hdp-2 has mesothoracic femur phenotype

(Montana and Littleton, 2004)

up¹⁰¹, wupA^hdp-2 has wing phenotype

(Nongthomba et al., 2003)

up¹⁰¹, wupA^hdp-2 has indirect flight muscle cell phenotype

(Nongthomba et al., 2003)

l(1)16Fe¹, wupA^hdp-2 has wing phenotype

(Prado et al., 1999)

l(1)16Fe¹, wupA^hdp-2 has adult somatic muscle cell phenotype

(Prado et al., 1999)

Additional Comments

Genetic Interactions

Statement

Reference

wupA^hdp-2/+ shows indirect flight muscle tearing in a trans-heterozygous condition with up¹⁰¹/+.

(Firdaus et al., 2015)

Galk^MB10638/+ fails to rescue the cardiac dilation and reduced fractional shortening phenotypes of wupA^hdp-2/+ mutants.

(Lee et al., 2014)

Expression of two copies of Ca-P60A^wt.tinC significantly rescues the cardiac function phenotype seen in wupA^hdp-2 mutant flies. Heart rate is still reduced.

(Abraham and Wolf, 2013)

Flight ability is largely rescues in flies carrying one copy of Act88F^D292V in a Act88F⁶/+ background if they are also hemizygous for wupA^hdp-2.

(Sevdali et al., 2013)

wupA^hdp-2; Mhc^D41/+ male adults show little defects in their indirect flight muscles; sarcomeres are almost normal, although some thin filaments bypass a Z disc and continue to the adjacent sarcomere.

(Marin et al., 2004)

Hemizygous flies for wupA^hdp-2 and heterozygous for Mhc^S1 or Mhc^S2 are synthetic lethal, whereas double heterozygous females have femur hypercontraction defects.

(Montana and Littleton, 2004)

The defects seen in wupA^hdp-2/Y flies are partially suppressed by Mhc¹⁰/+; 49% of the double mutant adults have an upheld wing

phenotype and 51% have their wings held down. 29% of the indirect

flight muscle fibres are hypercontracted, 71% show a partial hypercontraction

phenotype.

The defects seen in wupA^hdp-2/Y flies are partially suppressed

by Mhc^{R57-24.Act88F}/+; 51% of the double mutant adults have an

upheld wing phenotype, 39% have their wings held down and 10% hold

their wings in the normal position. 61% of the indirect flight muscle

fibres are hypercontracted, 39% show a partial hypercontraction phenotype.

The defects seen in wupA^hdp-2/Y flies are almost completely suppressed

by Mhc¹⁰/+ ; Mhc^{R57-24.Act88F}/+ - 13% of the double mutant

adults have an upheld wing phenotype, 0% have their wings held down

and 87% hold their wings in the normal position. 13% of the indirect

flight muscle fibres show a partial hypercontraction phenotype and

87% are normal.

The defects seen in wupA^hdp-2/Y flies are partially suppressed

by Mhc^2D/+; 49% of the double mutant adults have an upheld wing

phenotype and 51% hold their wings in the normal position. 10% of

the indirect flight muscle fibres are hypercontracted, 85% show a partial

hypercontraction phenotype and 5% are normal.

The wing and indirect flight muscle defects are completely suppressed

in wupA^hdp-2/Y ; Mhc^2D/Mhc^2D flies, although the sarcomeres

are still shorter than normal. For 6-7 days after eclosion the muscle

structure remains completely normal, but after this the central myofibrillar

lattice becomes disordered, with characteristic Z-band streaming and

gaps in the lattice.

The defects seen in wupA^hdp-2/Y flies are partially suppressed

by Mhc^2F/+; 28% of the double mutant adults have an upheld wing

phenotype and 72% hold their wings in the normal position. 11% of

the indirect flight muscle fibres show a partial hypercontraction phenotype

and 89% are normal. Sarcomere structure is comparable to wild type

in these flies, but sarcomere length is significantly shorter than

normal, and a few days after eclosion muscle fibres become thin in

many areas and contract.

Su(2)E^E is a very weak suppressor of the wing and muscle phenotype

of wupA^hdp-2.

wupA^hdp-2/Y ; Mhc^2B/+ flies show a complete recovery of wild-type

myofibrillar structure in the indirect flight muscles, although the

sarcomere length remains slightly, but significantly shorter than that

of wild type.

The defects seen in wupA^hdp-2 flies are suppressed by Mhc^D1/+;

7% of the double mutant adults have an upheld wing phenotype and 93%

hold their wings in the normal position. The indirect flight muscle

fibres have normal morphology.

The defects seen in wupA^hdp-2 flies are suppressed by Mhc^D41/+;

4% of the double mutant adults hold their wings down, 2% have an upheld

wing phenotype and 94% hold their wings in the normal position. The

indirect flight muscle fibres have normal morphology.

The defects seen in wupA^hdp-2 flies are suppressed by Mhc^D45/+;

7% of the double mutant adults have an upheld wing phenotype and 93%

hold their wings in the normal position. The indirect flight muscle

fibres have normal morphology.

The defects seen in wupA^hdp-2 flies are suppressed by Mhc^D62/+;

16% of the double mutant adults have an upheld wing phenotype and 84%

hold their wings in the normal position. The indirect flight muscle

fibres have normal morphology.

96% of up¹⁰¹ +/+ wupA^hdp-2 flies have an upheld wing phenotype

and 4% hold their wings down. The indirect flight muscle fibres show

a hypercontraction phenotype. 92% are flightless.

(Nongthomba et al., 2003)

The hypercontraction of indirect flight muscles seen in wupA^hdp-2 flies is completely suppressed by Tm2^D53/+. IFMs of 2-3 day old wupA^hdp-2 ; Tm2^D53/+ flies show an almost complete restoration of wild-type myofibrillar structure. However, some myofibrils show small areas of disorganised filament lattice at their centres. By 6-7 days, all of the myofibrils contain extensive disorganised areas. wupA^hdp-2 myofibrillar defects are completely suppressed by Tm2^D53/Tm2^D53. wupA^hdp-2 flies that also carry one or two copies of Tm2^D53 hold their wings in the resting position. Young wupA^hdp-2 ; Tm2^D53/+ or wupA^hdp-2 ; Tm2^D53/Tm2^D53 flies can fly, but less well than wild type. By 6-7 days old, the flight ability of wupA^hdp-2 ; Tm2^D53/+ but not wupA^hdp-2 ; Tm2^D53/Tm2^D53 flies is dramatically reduced. wupA^hdp-2 ; Tm2^D53/+ flies show some suppression of the wupA^hdp-2 leg muscle phenotype. The larval locomotory and feeding behaviour defects of wupA^hdp-2 larvae are suppressed by Tm2^D53. The upheld wing phenotype of wupA^hdp-2 flies is partially suppressed by Tm2^DL2/+ or Tm2^C10/+ but is not suppressed by Tm2^J8/+ or Tm2^L2/+. The IFM hypercontraction phenotype seen in wupA^hdp-2 flies is partially suppressed by Tm2^DL2/+, Df(3R)ea-5022rx1/+, Tm2^J8/+ or Tm2^C10/+ but is not suppressed by Tm2^L2/+.

(Naimi et al., 2001)

The DLM and heldout wing defects of wupA^hdp-2 flies are dominantly suppressed by Mhc^D1, Mhc^D41, Mhc^D45 or Mhc^D62. wupA^hdp-2/Y ; Mhc^D1/+ males have a near normal wing position, but cannot jump or fly. DLM fibres a to d appear almost normal in these flies, but DLM muscles e and f are collapsed. The number of thick filaments per fibril is reduced compared to wild type. wupA^hdp-2/Y ; Mhc^D41/+ males have a near normal wing position, but cannot jump or fly. DLM fibres a to d appear almost normal in these flies, but DLM muscles e and f have structural defects near the posterior end. The number of thick filaments per fibril is reduced compared to wild type. wupA^hdp-2/Y ; Mhc^D45/+ males have a near normal wing position, but cannot jump or fly. DLM fibres a to c appear almost normal in these flies, but DLM muscles e and f have structural defects near the posterior end, and muscle d also appears abnormal. The number of thick filaments per fibril is slightly reduced compared to wild type. The fibrils appear particularly unstable at the periphery, where the lattice collapses. wupA^hdp-2/Y ; Mhc^D62/+ males have a near normal wing position, but cannot jump or fly. DLM fibres a to d appear almost normal in these flies, but DLM muscles e and f have gross structural defects near the posterior attachment site. The number of thick filaments per fibril is reduced compared to wild type. wupA^hdp-2/Y ; Mhc^D1/Mhc^D41 flies can jump. wupA^hdp-2/Y ; Mhc^D1/Mhc^D62 flies have a high frequency of double thick filaments in the DLMs. Lethal in combination with Mhc⁵. The heldout wing defects of wupA^hdp-2 flies are not suppressed by Mhc⁶, Mhc⁸ or Df(2L)H20.

(Kronert et al., 1999)

l(1)16Fe¹ fails to complement wupA^hdp-2 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) fully complements wupA^hdp-2.

(Prado et al., 1999)

In double mutants with Mhc⁷ the flight muscles appear normal and do not degenerate.

(Beall and Fyrberg, 1991)

wupA^hdp-2 Mhc⁸ double heterozygotes are lethal. Female wupA^hdp-2 Mhc⁵ double heterozygotes are completely flightless and have an abnormal wing posture. Male wupA^hdp-2 heterozygotes also hemizygous for Mhc⁵ have reduced viability, the rare escapers have "gnarled" legs, walk poorly, and die within 2 days of eclosion. Double heterozygotes with Actn³, Actn¹⁴, Actn⁸ or Actn⁴ are flightless. Double heterozygotes with up^int-3, up¹⁰¹ or up^x are flightless and have an abnormal wing posture.