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To construct GMR-GAL4/Y; UAS-hDuox2/?, virgin female GMR-GAL4 flies containing two copies of the GMR-GAL4 gene on the X chromosome were mated with male hDuox2 flies containing two copies

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A Drosophila model for the screening of bioavailable NADPH

oxidase inhibitors and antioxidants

Nguyen Thi Tu Anh• Maiko Nishitani•

Shigeharu Harada• Masamitsu Yamaguchi•

Kaeko Kamei

Received: 16 October 2010 / Accepted: 28 January 2011 / Published online: 11 February 2011

Ó Springer Science+Business Media, LLC 2011

Abstract NADPH oxidase is the major source of

non-mitochondrial cellular reactive oxygen species (ROS), and

also is reported to be a major cause of various diseases

including atherosclerosis and hypertension In order to

screen a new curative reagent that can suppress NADPH

oxidase activity, we developed a Drosophila melanogaster

fly that would overexpress human Dual oxidase 2

(hDuox2), a member of the NADPH oxidase family, as a

screening model These flies (GMR-GAL4/UAS-hDuox2)

had a high generation of ROS in the posterior region of the

eye discs along with an easily recognizable rough-eye

phenotype, which is an ideal and convenient marker for

further screening steps Moreover, the hDuox2-induced

rough-eye phenotype can be rescued by feeding with a

culture medium containing mulberry leaves (MLs), which

reportedly have an antimetabolic effect Some

commer-cially available antioxidants such as quercetin-3-O-D

-glu-coside or quercetin-3-O-glucose-600-acetate, or the naringin

contained in MLs and other herbs, also have shown a

similar suppressing effect on the rough-eye phenotype Our

results suggest that flavonoid glycoside is absorbed from

the intestine and functions in the body of D melanogaster

as it does in mammalian models such as rats Thus, the

GMR-GAL4/UAS-hDuox2 fly line is a promising model

for the screening of novel drugs such as NADPH oxidase

inhibitors and/or antioxidants

Keywords NADPH oxidase  Drosophila melanogaster  Metabolic syndrome model  Screening  Antioxidant  NADPH oxidase inhibitor

Introduction Reactive oxygen species (ROS) play a critical role in the oxidative stress that causes various diseases, including atherosclerosis and hypertension [1 3] In recent years, it has become clear that ROS are produced by specific enzymes, including NADPH oxidase, by transferring one electron from NADPH to molecular oxygen [4] The NADPH oxidase family exists, with conserved functions,

in a wide variety of cell types from many organisms, including mammals, nematodes, fruit flies, green plants, fungi and slime molds [5] Inappropriate activation of NADPH enzymes may contribute to tissue dysfunction such as damage to the liver and pancreatic b-cells that leads to progression from the metabolic syndrome to Type

2 diabetes [6] Recently, Sugimoto et al discovered that inhibition of ROS generation via the suppression of NADPH oxidase expression in white adipose tissues and liver by the administration of mulberry leaves (MLs) ameliorated metabolic disorders in a mouse model [7] As well, several studies have revealed that MLs contain many antioxidants, particularly quercetin-3-(6-malonyglucoside), which is the most abundant flavonoid glycoside that has attenuated atherosclerotic lesion development in LDL receptor-deficient mice [8] These studies indicated that NADPH oxidase is an important target of antimetabolic drugs that may be present in MLs In order to screen new antimetabolic substances via the suppression of NADPH activity using MLs and other substances, the development

of a metabolic syndrome model is needed Drosophila

N T T Anh  M Nishitani  S Harada  M Yamaguchi 

K Kamei (&)

Department of Applied Biology, Kyoto Institute of Technology,

Matsugasaki Sakyo-ku, Kyoto 606-8585, Japan

e-mail: kame@kit.ac.jp

DOI 10.1007/s11010-011-0743-3

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melanogaster might be a viable candidate for studies of

metabolic syndrome because it shares most of the same

basic metabolic functions with vertebrates Many

analo-gous organ systems that control nutrient uptake, storage

and metabolism in humans are present in fruit flies [9] In

addition, their rapid growth, inexpensive costs of breeding,

and small genome size facilitates screening steps

Human dual oxidases (hDuox1 and hDuox2), members of

the NADPH oxidase family, are expressed in the thyroid, the

airway epithelial cells and in a wide variety of tissues such as

brain, cerebellum, mammary gland, muscle, nervous,

pla-centa, and testis The genes encoding for these two enzymes

are located on chromosome 15, and share 83% sequence

similarity [10] In the thyroid gland, Duox enzymes produce

hydrogen peroxide, which is essential for the oxidation step

of iodide in thyroid hormone production The important role

of Duox2 in this process was proven by the identification of

patients who suffer from hypothyroidism due to mutations in

the Duox2 gene [11] Besides, both in human and other

organisms, dual oxidases are novel H2O2sources that are

indispensable in host defense against infection and

extra-cellular matrix modification [12–14]

Recently, the gut antimicrobial activities of flies, in

which Drosophila NADPH oxidase (dDuox) had been

silenced, could be markedly ameliorated by the

introduc-tion of the hDuox2 gene, indicating that hDuox2 is

func-tional in Drosophila [13] Therefore, in order to construct

an in vivo screening system for new antimetabolic

sub-stances via the suppression of NADPH activity, we utilized

the GAL4-UAS expression system to provide ectopic

expression of hDuox2 in D melanogaster [15] GAL4 is a

yeast transcriptional activator that can activate the

tran-scription of any gene after its introduction into

Drosoph-ila—if the gene is preceded by a GAL4 upstream activating

sequence (UAS) that consists of five optimized GAL4

binding sites [16] In this study, a transgenic fly with a

GMR (glass multiple reporter)-GAL4 gene on chromosome

X was crossed with a fly with a UAS-hDuox2 gene on

chromosome 2 Since the GMR element causes high-level

expression of GAL4 protein in cells from the posterior to

the morphogenetic furrow, we were able to obtain an

expression of hDuox2 in the posterior area of the eye discs

This system has the advantage of allowing evaluation of

hDuox2 activity by simple inspection of the eye phenotype

Furthermore, we evaluated the use of flies that

overex-press hDuox2 as models for the screening of antimetabolic

substances The evaluation was carried out by feeding MLs

and their flavonoids to transgenic Drosophila Mulberry

leaves are reported to have an antimetabolic effect due to

their content of flavonoids with antioxidant activities The

effectiveness of MLs and these substances was evaluated

based on their ability to suppress the hDuox2-induced

rough-eye phenotype

Materials and methods Materials

Quercetin was purchased from Nacalai Tesque (Kyoto, Japan) The rutin, epigallocatechin gallate, and naringin were acquired from Wako Pure Chemical (Osaka, Japan) Quercetin-3-O-D-glucoside, quercetin-3-O-glucose-600 -ace-tate, and kaempherol all were acquired from Extrasynthese (France)

Mulberry leaves The mulberry trees were cultured in a mulberry plantation

at the Center for Bioresource Field Science, Kyoto Institute

of Technology, using a standard Japanese method The

‘‘Shin-Ichinose’’ mulberry (Morus alba L.) race was used Mulberry leaves were harvested and immediately dried by air flush at 180°C for 7 s The average diameter of the dried powder was 20 lm

Fly stocks Fly stocks were maintained at 25°C The UAS-hDuox2 fly line that carries the human Duox2 gene on second chromo-some was kindly provided by Dr Won-Jae Lee, of Ewha Woman’s University, South Korea [13] The GMR-GAL4/ UAS-DREF line was used as a control strain [17] The transgenic fly line carrying GMR-GAL4 on the X chromo-some (strain number 16) has been described previously [17] Establishment of transgenic flies

Virgin females were used for every crossing step to insure purity

To construct GMR-GAL4/Y; UAS-hDuox2/?, virgin female GMR-GAL4 flies containing two copies of the GMR-GAL4 gene on the X chromosome were mated with male hDuox2 flies containing two copies of the UAS-hDuox2 gene on the second chromosome In the next generation, all male flies carry a single copy of both GMR-GAL4 and hDuox2 (GMR-GMR-GAL4/Y; UAS-hDuox2/?)

To construct the GMR-GAL4; UAS-hDuox2 line, virgin female GMR-GAL4 flies containing two copies of the GMR-GAL4 gene on the X chromosome and a second-balancer chromosome CyO (GMR-GAL4; Sp/CyO) were crossed with male UAS-hDuox2 flies containing two cop-ies of the UAS-hDuox2 gene on the second chromosome The second chromosome balancer CyO suppresses recombination with their homologs, carries dominant markers (curly-wing phenotype), and causes a lethal effect when it is homozygous In the next generation, all male

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flies surely contain one copy of GMR-GAL4 on the X

chromosome These male flies were further selected by

curly-wing phenotype to get one copy of UAS-hDuox2 and

one second chromosome balancer, CyO (GMR-GAL4/Y;

UAS-hDuox2/CyO) These flies crossed again with female

GMR-GAL4; Sp/CyO Subsequently, we could select

female GAL4; UAS-hDuox2/CyO and male

GMR-GAL4/Y; UAS-hDuox2/CyO These two fly lines were

mated together to finally establish fly lines containing two

copies of GMR-GAL4 on the X chromosome and two

copies of UAS-hDuox2 on the second chromosome

(GMR-GAL4; UAS-hDuox2)

Culture medium

The medium buffer contained 1% yeast extract and 0.5%

agar in phosphate-buffered saline (PBS)

ML-supple-mented culture medium for feeding was prepared by

mix-ing 750 ll medium buffer with 0.15 g instant medium

Formula 4–24Ò (Carolina Biological Supply Co., USA)

and 10, 20, or 30% (w/w) of MLs Antioxidant stocks were

prepared by being dissolved in 99% ethanol

Antioxidant-supplemented mediums were prepared by dissolving

anti-oxidant stocks into medium buffer before mixing with

instant medium Formula 4–24Òto obtain a final 3% (w/w)

concentration A blue coloring agent contained in the

instant medium facilitated recognition of whether the

lar-vae consumed the tested medium

Feeding method

Three males and three females of the control (GMR-GAL4;

UAS-DREF) or the hDuox2-overexpressing flies

(GMR-GAL4; UAS-hDuox2) were, respectively, mated to lay

eggs on culture mediums containing various concentrations

of mulberry powder or antioxidants for 1 day Newborn

larvae were continuously fed on the same mediums for

10 days at 25°C to reach the adult phase

Scanning electron microscopy

Adult flies were anesthetized, mounted on stages, and

observed using a VE-7800 (Keyence Inc., Osaka, Japan)

scanning electron microscope in high vacuum mode In

every experiment, the eye phenotype of at least five adult

flies of each line was simultaneously examined by scanning

electron microscopy, and these experiments were repeated

three times In the experiments, no significant variation in

eye phenotype among the five individuals was observed

The rough area of the eyes was circled as an index of the

effect on the rough-eye phenotype

In vivo ROS detection Eye discs from third instar larvae were dissected in PBS, and then incubated with 10 lM CM-H2DCFDA (5-(and-6)-carboxy-20, 70-dichlorodihydrofluorescein diacetate, acetyl ester) (Molecular Probes, Invitrogen) for 5 min After washing with PBS, samples were fixed in 1% para-formaldehyde for 5 min, washed three times with PBS, and then mounted in Vectashield Mounting Medium (Vector laboratories) Preparations were examined under a fluo-rescence BX-50 microscope (Olympus, Tokyo, Japan) equipped with a cooled CCD camera (ORCA-ER; Ham-amatsu Photonics K.K., Shizuoka, Japan)

Immunohistochemistry Third instar larvae were dissected in PBS and eye discs were fixed in 4% paraformaldehyde–PBS for 30 min at 25°C After washing with 0.3% Triton X-100 in PBS (PBST), samples were blocked for 30 min at 25°C with 0.15% PBST containing 1% bovine serum albumin Sam-ples were then incubated with rabbit anti-hDuox2 antibody (Abcam) at a 1:100 dilution and 4°C for 16 h After extensive washing with PBST, the samples were incubated with goat anti-rabbit IgG Alexa FluorTM 488 (Molecular Probes, Invitrogen) at a 1:400 dilution for 2 h at 25°C, further washed with PBST and PBS, then mounted in Vectashield Mounting Medium (Vector laboratories) Preparations were examined under a fluorescence BX-50 microscope (Olympus, Tokyo, Japan) equipped with a cooled CCD camera (ORCA-ER; Hamamatsu Photonics K.K., Shizuoka, Japan)

Results Overexpression of human Duox2 disrupted normal eye development

The hDuox2 transgenic flies were successfully used to rescue the defect in antimicrobial infection activity in the Drosophila NADPH oxidase (dDuox) RNAi strain, indi-cating that hDuox2 is functional in Drosophila [13] In this study, we utilized the GMR-GAL4 driver strain to provide ectopic expression of hDuox2 from the posterior to the morphogenetic furrow of a Drosophila eye [17] Under scanning electron microscope, the eyes of flies carrying a single copy of GMR-GAL4 (GMR-GAL4/Y; ?) appeared

as a normal phenotype (Fig 1a), while those of flies car-rying a single copy of both GMR-GAL4 and hDuox2 (GMR-GAL4/Y; UAS-hDuox2/?) had a mild rough-eye phenotype (Fig.1b) When the copy number of both GMR-GAL4 and hDuox2 were increased to two (GMR-GMR-GAL4;

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UAS-hDuox2, Fig.1d), the compound eyes of these flies

had many fused ommatidia and became more severely

rough than those of flies containing two copies of

GMR-GAL4 only (GMR-GMR-GAL4; ?, Fig.1c)

To further confirm the overexpression of hDuox2 in the

compound eyes, we performed immunostaining of eye

imaginal discs from third instar larvae using anti-hDuox2

polyclonal antibody Only in GMR-GAL4; UAS-hDuox2,

an ectopic hDuox2 signal from the posterior region to the

morphogenetic furrow in eye discs was detected (Fig.2b)

The GMR-GAL4 line that was used as the negative control

showed no signal (Fig.2a) Immunostaining of eye

imag-inal discs of both GMR-GAL4; ? and GMR-GAL4;

UAS-hDuox2 using only goat anti-rabbit IgG Alexa FluorTM488

were also carried out to further exclude background effects

(Fig.2c, d, respectively)

Overexpression of hDuox2 increased ROS generation

Overexpression of hDuox2 can lead to an increased

gen-eration of ROS To detect intracellular ROS in

hDuox2-overexpressing flies, we used the non-fluorescent substrate

CM-H2DCFDA, which can be oxidized by ROS to become

an intracellular green fluorescent product With no

treat-ment with substrate CM-H2DCFDA, no detectable signal

was observed in eye imaginal discs for either control flies

GMR-GAL4 or hDuox2-overexpressing flies, as shown in Fig.3a and b, respectively In contrast, upon treatment with substrate CM-H2DCFDA, hDuox2-overexpressing flies showed stronger fluorescent signals from the posterior region to the morphogenetic furrow in eye discs (Fig.3d)

as compared with that of the control GMR-GAL4 fly line (Fig.3c) This indicates that ROS generation was induced

by the overexpression of hDuox2 in eye discs

Mulberry leaves could suppress the hDuox2-induced rough-eye phenotype

In previous research, metabolic disorder was ameliorated in obese db/db mice fed MLs, at least partially by the sup-pression of oxidative stress by both antioxidative flavonoid scavenging of ROS and by the inhibition of NADPH oxi-dase expression [7,18] Therefore, in this study, to evaluate the use of GMR-GAL4/UAS-hDuox2 as a metabolic syn-drome model, we fed these flies 10, 20, and 30% (w/w) MLs To exclude effects of MLs other than on hDuox2 activity, flies overexpressing DREF, a transcription factor,

by GMR-GAL4 with approximate rough-eye phenotype were used as a control [17] As shown in Fig.4a–d, MLs had no effect on the eye phenotype of DREF-over-expressing flies, indicating that MLs exerted no effect on GAL4 expression However, the rough-eye phenotype of

Fig 1 Adult compound eyes observed by scanning electron

micros-copy a The eyes of flies carrying a single copy of GMR-GAL4

(GMR-GAL4/Y; ?) appeared as a normal phenotype; b flies carrying

a single copy of both GMR-GAL4 and hDuox2 (GMR-GAL4/Y;

UAS-hDuox2/?) showed a mild rough-eye phenotype; c flies

carrying two copies of GMR-GAL4 (GMR-GAL4; ?) had many fused ommatidia; d flies carrying two copies of both GMR-GAL4 and hDuox2 (GMR-GAL4; UAS-hDuox2) showed a more severe rough-eyes phenotype Magnification is 2009 for left panels and 7009 for right panels Flies were reared at 25°C

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GMR-GAL4/UAS-hDuox2 was suppressed effectively

when feeding with a culture medium containing 20% MLs

(Fig.4g) The extent of suppression was more increased as

the concentration of MLs was increased up to 30% in the

medium (Fig.4h) The suppressive effects can be realized

by comparing the rough areas of eyes marked by dot circles

in Fig.4 These results suggest that the

GMR-GAL4/UAS-hDuox2 fly is a potential model for the screening of new

antimetabolic substances

Antioxidants could suppress the hDuox2-induced

rough-eye phenotype

Some phenolic compounds that have antioxidative

activi-ties, including quercetin, rutin, kaempherol,

epigallocate-chin gallate, quercetin-3-O-D-glucoside,

quercetin-3-O-glucose-600-acetate, and naringin, were tested for the ability

to suppress the hDuox2-induced rough-eye phenotype The oral administration of quercetin, rutin, kaempherol, and epigallocatechin gallate did not affect the rough-eye phenotype of hDuox2-overexpressing flies (Fig.5b–e) However, flavonoid glycosides such as quercetin-3-O-D -glucoside, quercetin-3-O-glucose-600-acetate, and naringin could significantly suppress the hDuox2-induced rough-eye phenotype (Fig.5f–h)

Discussion

In this study, we crossed UAS-hDuox2 with GMR-GAL4 driver to establish a fly line overexpressing hDuox2 The eyes of these flies appeared severely rough compared with

Fig 2 Immunostaining of eye imaginal discs with anti-hDuox2

antibody Eye imaginal discs were reacted with rabbit anti-hDuox2

antibody followed by anti-rabbit IgG Alexa FlourTM 488 antibody

(a, b) a Immunostaining of eye discs of GMR-GAL4; ? showed no

detectable signal; b notably, in GMR-GAL4; UAS-hDuox2, an

ectopic hDuox2 signal from the posterior region to the morphogenetic

furrow in eye discs was detected Immunostaining of eye imaginal discs of both GMR-GAL4; ? (c) and GMR-GAL4; UAS-hDuox2 (d) with only the anti-rabbit IgG Alexa FlourTM488 antibody showed

no detectable signal Flies were reared at 28°C The arrowheads indicate morphogenetic furrows in the eye discs

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Fig 3 Detection of ROS in eye

imaginal discs of third instar

larvae Without substrate

CM-H2DCFDA, there were no

detectable signals in either

GAL4; ? (a) or

GMR-GAL4; UAS-hDuox2 flies (b).

However, by incubation with

substrate CM-H2DCFDA, a

weak fluorescent signal in

control flies (GMR-GAL4; ?)

(c) was detected Remarkably, a

strong fluorescent signal was

detected in

hDuox2-overexpressing flies

(GMR-GAL4; UAS-hDuox2) (d) Flies

were reared at 25°C The

posterior of the discs are on the

right The arrowheads indicate

morphogenetic furrows in the

eye discs

Fig 4 Effect of MLs on the hDuox2-induced rough-eye phenotype.

Scanning electron micrographs of adult compound eyes at 2009

magnification of GMR-GAL4; UAS-DREF (a–d) and GMR-GAL4;

UAS-hDuox2 (e–h) GMR-GAL4; UAS-DREF flies fed culture

mediums containing 10% (b), 20% (c), or 30% (d) MLs showed no

effect on the rough-eye phenotype as compared to flies not fed MLs

(a) However, compared to flies not fed MLs (e), the rough-eye phenotype of hDuox2-overexpressing flies was suppressed when fed culture mediums containing 10% (f), 20% (g), or 30% (h) MLs The suppressive effects can be realized by comparing rough areas of the eyes marked by dot circles Flies were reared at 25°C

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GMR-GAL4 flies By immunohistochemical analysis with

anti-hDuox2 antibody, we proved that these flies expressed

an abundant amount of hDuox2 protein in the posterior

region to the morphogenetic furrow in eye discs

Subse-quently, the production of ROS in this region also

increased indicating that the hDuox2 enzyme functioned in

these flies These results showed that

GMR-GAL4/UAS-hDuox2 flies induced high oxidative stress in the posterior

region of the eye discs that led to an easily recognizable

rough-eye phenotype in adults

We continued to feed this fly line with MLs and several

antioxidative flavonoids to evaluate the ability of the line to

be used as a model for the screening of antimetabolic

substances The results show that, with increasing

con-centration of MLs in the culture medium, the suppressive

effect of MLs became more prominent Similar results

were obtained when culture medium containing 3% (w/w)

flavonoid glycoside, quercetin-3-O-D-glucoside,

quercetin-3-O-glucose-600-acetate, or naringin was fed to the fly

However, it is noteworthy that flavonoids without a

glycosidic ring, quercetin, rutin, kaempherol, and epigal-locatechin gallate exerted no effect on the hDuox2-induced rough-eye phenotype This differential effect could be explained by the differences between flavonoids and their glycosides in intestinal absorption and bioavailability due

to chemical structures and water solubility raised from attached glycoside rings that had been described in previ-ous human and rat studies [19–21] Our results might indicate that the absorption of antioxidants in the intestine

of D melanogaster is similar to mammalian models such

as rats

In this study, we used flavonoids as ROS scavengers to evaluate the fly as a screening tool, and, as well, the transgenic fly can be used to screen for NADPH oxidase inhibitors However, our screening system cannot distin-guish between ROS scavengers and NADPH oxidase inhibitors Therefore, further ROS scavenging assay using chemicals and/or kinetic assay of ROS generation by NADPH oxidase should be carried out Despite this limi-tation, Drosophila has many advantages as a screening

Fig 5 Effect of flavonoids on the hDuox2-induced rough-eye

phenotype Scanning electron micrographs of adult compound eyes

at 2009 magnification of GMR-GAL4; UAS-hDuox2 Untreated

GMR-GAL4; UAS-hDuox2 flies showed a severe rough-eye

pheno-type (a) Treatment with 3% (w/w) quercetin (b), rutin (c),

kaempherol (d), and epigallocatechin gallate (e) showed no detectable changes in compound eyes However, treatment with 3% (w/w) quercetin-3-O- D -glucoside (f), quercetin-3-O-glucose-6 00 -acetate (g), and naringin (h) strongly suppressed the hDuox2-induced rough-eye phenotype Flies were reared at 25°C

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system for new drugs Drosophila is easier to

inexpen-sively rear in the laboratory, has a shorter generation time,

and produces much more progenies when compared with

mice or rats Therefore, the use of our model is

recom-mended for the first simultaneous screenings of a great

number of samples In addition, because of the similar

basic metabolic functions and analogous organs between

Drosophila and vertebrates [9], the

GMR-GAL4/UAS-hDuox2 fly line constructed in our study is a promising

model for the screening of bioavailable novel drugs

Acknowledgments We are grateful to Dr Won-Jae Lee, Ewha

Woman’s University, South Korea for the UAS-hDuox2 stock.

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