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Thioredoxin reductase from the malaria mosquitoAnopheles gambiae Comparisons with the orthologous enzymes of Plasmodium falciparum and the human host Holger Bauer1, Stephan Gromer1, Andr

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Thioredoxin reductase from the malaria mosquito

Anopheles gambiae

Comparisons with the orthologous enzymes of Plasmodium falciparum

and the human host

Holger Bauer1, Stephan Gromer1, Andrea Urbani2, Martina Schno¨lzer2, R Heiner Schirmer1

and Hans-Michael Mu¨ller3

1

Biochemie Zentrum, Universita¨t Heidelberg, Heidelberg, Germany;2Deutsches Krebsforschungszentrum, Heidelberg, Germany;

3

European Molecular Biology Laboratory, Heidelberg, Germany

The mosquito, Anopheles gambiae, is an important vector of

Plasmodium falciparum malaria Full genome analysis

revealed that, as in Drosophila melanogaster, the enzyme

glutathione reductase is absent in A gambiae and

func-tionally substituted by the thioredoxin system The key

enzyme of this system is thioredoxin reductase-1, a

homo-dimeric FAD-containing protein of 55.3 kDa per subunit,

which catalyses the reaction NADPH + H++

thio-redoxin disulfidefi NADP++ thioredoxin dithiol The

A gambiae trxr gene is located on chromosome X as a

single copy; it represents three splice variants coding for two

cytosolic and one mitochondrial variant The predominant

isoform, A gambiae thioredoxin reductase-1, was

recomb-inantly expressed in Escherichia coli and functionally

com-pared with the wild-type enzyme isolated in a final yield

of 1.4 UÆml)1 of packed insect cells In redox titrations,

the substrate A gambiae thioredoxin-1 (Km¼ 8.5 lM,

kcat¼ 15.4 s)1at pH 7.4 and 25C) was unable to oxidize

NADPH-reduced A gambiae thioredoxin reductase-1 to

the fully oxidized state This indicates that, in contrast to

other disulfide reductases, A gambiae thioredoxin

reduc-tase-1 oscillates during catalysis between the four-electron reduced state and a two-electron reduced state The thio-redoxin reductases of the malaria system were compared

A gambiaethioredoxin reductase-1 shares 52% and 45% sequence identity with its orthologues from humans and

P falciparum,respectively A major difference among the three enzymes is the structure of the C-terminal redox cen-tre, reflected in the varying resistance of catalytic inter-mediates to autoxidation The relevant sequences of this centre are Thr–Cys–Cys–SerOH in A gambiae thioredoxin reductase, Gly–Cys–selenocysteine–GlyOH in human thio-redoxin reductase, and Cys–X–X–X–X–Cys–GlyOH in the

P falciparumenzyme These differences offer an interesting approach to the design of species-specific inhibitors Notably, A gambiae thioredoxin reductase-1 is not a selenoenzyme but instead contains a highly unusual redox-active Cys–Cys sequence

Keywords: Anopheles gambiae; Drosophila melanogaster; Diptera; insect redox metabolism; Plasmodium falciparum

The mosquito Anopheles gambiae is of importance as a

vector of tropical malaria caused by the protozoan organism

Plasmodium falciparum The genome of A gambiae is the

second insect genome – after the distantly related model

organism Drosophila melanogaster [1,2] – that has been completely sequenced [3] Annotation of the nucleotide sequences allows access to the genetic background of a disease-transmitting dipteran insect and, furthermore, offers the opportunity of comparative sequence analyses from single genes up to genomic organization [4] Another aspect is highlighted in this report: only on the basis of the full genome sequence does it become possible to exclude the presence of a given protein function in all cells and all developmental stages

of an organism A case in point is the absence of the enzyme glutathione reductase (GR) in Diptera [5]

Our focus is the redox metabolism of insects [6] Being present at millimolar concentrations, the tripeptide gluta-thione (GSH) is the most abundant antioxidative thiol compound in most cell compartments Its redox state determines the intracellular redox environment [7] Thus, GSH is the major redox buffer compound and essential for the detoxification of free radicals and xenobiotics [8,9]

In the majority of pro- and eukaryotic organisms, the oxidized form of glutathione (glutathione disulfide, GSSG) is reduced to the mono-thiol form (GSH) by the

Correspondence to R H Schirmer, Biochemie Zentrum, Im

Neuen-heimer Feld 504, D-69120 Heidelberg, Germany.

Fax: + 49 6221 545586, Tel.: + 49 6221 544165,

E-mail: heiner.schirmer@gmx.de or H.-M Mu¨ller, European

Molecular Biology Laboratory, Meyerhofstr 1, D-69117 Heidelberg,

Germany Fax: + 49 6221 387306, Tel.: + 49 6221 387440,

E-mail: hmueller@embl-heidelberg.de

Abbreviations: EH 2 , enzyme in a two-electron reduced state; EH 4 ,

enzyme in a four-electron reduced state; E ox , enzyme in an oxidized

state; EST, expressed sequence tag; GR, glutathione reductase;

GSH/GSSG, reduced/oxidized glutathione; Sec, selenocysteine;

Trx, thioredoxin; TrxR, thioredoxin reductase.

(Received 14 July 2003, revised 29 August 2003,

accepted 1 September 2003)

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NADPH-dependent flavoenzyme GR, which catalyses the

following reaction: NADPH + GSSG + H+fi NADP++

2GSH [10–12] D melanogaster cells exhibit a high 2[GSH]/

[GSSG] ratio but have been shown to lacka typical GR [5]

As reported here, this is also true for A gambiae An

important candidate able to functionally substitute for

GR is the thioredoxin (Trx) system [13], which

compri-ses NADPH, thioredoxin reductase(s) (TrxR) and Trxs

[14,15]

Trxs are small, ubiquitous thiol proteins with a relative

molecular mass of 12 kDa and a redox active cysteine

pair represented in a WCGPC sequence motif Therefore,

they cycle between a disulfide (TrxS2) and a dithiol

[Trx(SH)2] form

Trxs were first described as electron-donating substrates

for ribonucleotide reductase [16], but they cleave disulfide

bonds in a number of other proteins equally well Thus, Trxs

take part in the redox control of numerous processes such

as protein folding, signalling and transcription [14,17–19]

Trx reduction is catalysed by the flavin-dependent

oxido-reductase TrxR, as follows: NADPH + TrxS2+ H+fi

NADP++ Trx(SH)2 TrxRs belong to a disulfide

reduc-tase superfamily that includes enzymes such as GR,

trypanothione reductase, lipoamide dehydrogenase, and

mercuric ion reductase These homodimeric proteins are

structurally, as well as mechanistically, closely related [20]

Evolution has produced two classes of TrxRs: small

TrxRs (found in bacteria, plants and fungi) and large

TrxRs (present in other eukaryotes) [21] In contrast to

GRs and low molecular weight TrxRs, large TrxRs

possess an additional redox centre located in the

C-terminal extension which is necessary for the interaction

with the substrate Trx In mammalian enzymes this redox

centre is represented by a neighboring

cysteine–seleno-cysteine pair [22] and in the TrxR of P falciparum it is a

cysteine pair separated by a spacer sequence of four

amino acids [23] D melanogaster TrxR was described as

the first member of a third type of large TrxRs It is

characterized by two adjacent cysteines preceding the

C-terminus [5,24]

High molecular mass TrxRs exhibit a rather broad

substrate spectrum that includes a number of natural and

also artificial disulfide compounds, such as

5,5¢-dithiobis(2-nitrobenzoate) Glutathione disulfide, however, is not a

substrate In the fruit fly it was shown that GSSG reduction

can occur in a dithiol–disulfide exchange reaction

with reduced Trx [5] At physiological concentrations of

GSSG and Trx, this system allows GSSG fluxes of

> 100 lMÆmin)1[25]

In this report we introduce the TrxR of the malaria

mosquito A gambiae The protein could be isolated from

whole insects and from cultured insect cells With the

progress of the Anopheles genome project it was possible to

identify the complete sequence of the gene and its

organi-zation We cloned, recombinantly expressed and

character-ized the enzyme Our data support the assumption that the

substitution of the Trx system for GR, as well as the

mechanistic particularities of the TrxR, are a common

principle in dipteran insects In the context of the malarial

system, this implies that the TrxRs of insect vector, parasite,

and human host differ in their cellular roles as well as their

enzyme mechanisms

Experimental procedures

D melanogasterTrx-2, A gambiae Trx-1 and P falciparum Trx-1 were prepared and purified as previously described [25,26] PCR chemicals and restriction enzymes were purchased from MBI Fermentas and Applied Biosystems, precast polyacrylamide gels from Bio-Rad and molecular mass standards from Amersham Pharmacia Biotech Anti-biotics, substrates for enzyme assays, and other chemicals were from BioMol, Fluka, or Sigma All compounds were

of the highest available purity

Purification of authenticA gambiae TrxR from insect cells

A gambiae cells (cell line 4a-2s4) were cultured in Schneider’s medium and harvested as described previ-ously [27] A 0.3-mL volume of lysis buffer (50 mMTris/ HCl, 3 mM EDTA, 2.5 mM phenylmethanesulfonyl fluoride, 5 lM pepstatin and 5 lM cystatin, pH 7.6) was added per mL of frozen cell pellet The pellets were thawed at 37C in a water bath, fresh phenyl-methanesulfonyl fluoride (ad 500 lM) was added, and the cells were disintegrated by ultrasound All subsequent steps were carried out at 4C The suspension was centrifuged for 1 h at 26 000 g The supernatant was set aside, and the pellet was resuspended in lysis buffer and centrifuged as described above The combined supernatants were mixed with two volumes of TE buffer (50 mMTris/HCl,

1 mMEDTA, pH 7.6) and slowly loaded onto a cooled 2¢,5¢-ADP–Sepharose column (1.5 mL per 10 mL of cell pellet) equilibrated with TE buffer The column was washed with two column volumes of TE buffer, 1.5 column volumes of

100 mMKCl in TE buffer, three column volumes of 1 : 3 diluted TE buffer, 1.5 column volumes of 1 mMNADH in

1 : 3 diluted TE buffer, and two column volumes of TE buffer TrxR activity was then eluted with 2 mMNADP+in

TE buffer Fractions containing significant amounts of activity were pooled, concentrated and washed with TE buffer in a 10-kDa Amicon concentrator Purity was analyzed by SDS/PAGE and silver staining GR activity was not detected in the crude extract or in any column fraction, even when the column was washed with 1MKCl and 1 mM NADPH in TE buffer The combined and concentrated washing solutions were stored at)80 C as a source of other NADPH-dependent enzymes of A gambiae TrxR assay

TrxR assays were conducted at 25C with a reaction volume

of 1 mL consisting of buffer T (100 mM potassium phos-phate, 2 mM EDTA, pH 7.4) and 100 lM NADPH For determination of the Kmvalues of Trxs, Trx concentrations were varied from 3 to 50 lM[25] The assays were started

by the addition of 10 milliunits of A gambiae TrxR-1 (1 unit¼ 1 lmol of NADPH consumption per minute under substrate saturation), and the Trx-dependent NADPH oxidation was followed spectrophotometrically at 340 nm applying an e-value of 6.22 mM )1Æcm)1 Kmand Vmaxvalues were obtained by applying the Michaelis–Menten equation

GR activity was determined using established protocols [28,29]

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Trx-dependent GSSG-reduction assay

The Trx-dependent GSSG-reduction assay was conducted

as described previously [25,26] The mixture contained

100 lM NADPH and A gambiae Trx-1 in concentrations

from 5 to 50 lM The assay was started by the addition of

1 U of A gambiae TrxR-1, and NADPH oxidation was

followed at 340 nm After reduction of Trx was complete,

1 mMGSSG was added, and GSSG reduction was followed

by further NADPH consumption The composition of the

assay mixture guarantees that > 98% of Trx is present in

the reduced form

L-Dehydroascorbate reduction assay for TrxR

L-Dehydroascorbate (dimer; Sigma-Aldrich) was studied as

a substrate in the range of 50 lMto 5 mMin TrxR assay

mixture containing 200 lMNADPH and 300 nMA

gamb-iaeTrxR subunits; NADPH consumption was determined

spectroscopically at 340 nm Purified human TrxR served

as a positive control

Protein determination

The protein concentration in crude fractions was estimated

assuming an absorption of 10 at 280 nm for a 1% solution

For determining the exact concentration of TrxR, flavin

absorbance was measured at 450 nm after denaturation

of the enzyme sample by 0.1% SDS and heating to 80C;

the FAD released by this procedure has an e-value of

11.3 mM )1Æcm)1

Sequence studies on authenticA gambiae TrxR

A 10 lg sample of purified authentic A gambiae TrxR was

applied per lane in reducing SDS/PAGE After

electro-phoresis, one lane was Coomassie-stained and the putative

TrxR band was subjected to tryptic digestion (see below)

Another lane was blotted [in 50 mM sodium borate, 20%

(v/v) methanol, pH 9.0, at 150 mA] overnight onto a

poly(vinylidene difluoride) membrane and stained with

0.1% amido-blackin 2% acetic acid This yielded a protein

band of 58 kDa An additional minor band of 62 kDa

appeared when phenylmethanesulfonyl fluoride was present

during all steps of the protein isolation Both bands were

excised and subjected to Edman degradation

Selenium analysis

Ten microgram samples of purified authentic A gambiae

TrxR were subjected to atomic absorption spectrometry for

selenium determination (Dr Muntean, Labor Seelig,

Karls-ruhe, Germany) A negative control (TE buffer) and a

positive control (10 lg of human TrxR in TE buffer) were

analysed in parallel

Cloning ofA gambiae TrxR-1

The A gambiae trxr-1 gene was PCR cloned from genomic

DNA as well as from the cDNA of adult insects In the case

of the amplification from genomic DNA, the bases coding

for the first five amino acids, located on the first exon, were

included in the primer For the cloning of genomic DNA, the following primers were applied: forward, 5¢-CGCAG GATCCGCGCCATTGAATCAGGAAAACTATGAGT ACGATCTGGTG-3¢ (containing a BamHI restriction site); and reverse, 5¢-TCCTAAGCTTCTAGCTGCAG CAGGTCGCCGGCGTCG-3¢ (containing a HindIII restriction site) Dimethylsulfoxide [5% (v/v)] was added

to the PCR mixture to improve amplification of the GC-rich gene PCR conditions were as follows: 94C for 60 s; 35 cycles of 30 s at 94C, 30 s at 68 C and 90 s at 72 C; then

10 min at 72C The PCR fragment was cloned into the expression vector, pQE-60 (Qiagen), and Escherichia coli NovaBlue cells (Novagen) were transformed with the plasmid The insert was verified by sequencing

Protein expression Transformed E coli NovaBlue cells were grown overnight

at 34C in 2· YT medium containing 50 lgÆmL)1 carbeni-cillin A gambiae TrxR-1 expression was then induced with 0.3 mM isopropyl-b-D-thiogalactopyranoside for 4 h at

34C After centrifugation (3000 g, 10 min, 4 C), cells were resuspended in 25 mM TE buffer and treated with lysozyme (0.2 mgÆmL)1) and DNase (0.02 mgÆmL)1) for

20 min at room temperature Phenylmethanesulfonyl fluo-ride (100 lM), pepstatin (3 lM) and cystatin (80 nM) were added as protease inhibitors and the cells were disintegrated

by ultrasound The homogenate was centrifuged at 38 000 g for 30 min at 4C and the supernatant was applied to a 2¢,5¢-ADP–Sepharose column equilibrated with 50 mMTE buffer The column was washed with five volumes of 25 mM

TE buffer and one volume of 50 mMTE buffer A gambiae TrxR-1 was eluted by 2 mM NADP+in 50 mM TE, the final yield being  40 mgÆL)1 of protein culture SDS/ PAGE, using a 10% gel, showed a single band of the expected size, the purity being > 95% as judged by silver staining

MS of tryptic peptides Protein bands were excised from SDS/PAGE, and their cysteine residues were reduced and alkylated with iodoacet-amide The samples were then digested with porcine trypsin (Promega) in 40 mMammonium bicarbonate at 37C for 6–8 h The reaction was stopped by freezing Tryptic peptides were extracted by ZipTip C18 reverse phase material (Millipore), chromatographed, and taken up in a saturated solution of a-cyano-4-hydroxycinnamonic acid in 50% (v/v) acetonitrile/water

MALDI mass spectra were recorded in the positive ion mode with delayed extraction on a Reflex IV time-of-flight instrument equipped with an MTP multiprobe inlet and a 337-nm nitrogen laser Mass spectra were obtained by averaging 50–200 individual laser shots Calibration of the spectra was internally performed by a two-point linear fit using the autolysis products of trypsin at m/z 842.50 and m/z 2211.10

The peptide masses were screened against the NCBInr database using the peptide search algorithm MASCOT

(Matrix Science) Fragments generated by postsource decay experiments were analysed using the database search algorithm - (http://prospector.ucsf.edu)

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When this project started, only one Trx (A gambiae Trx-1)

had been described as a part of the Trx-based redox

metabolism in the malaria mosquito A gambiae [25]

Complementary studies on the fruit fly, D melanogaster

[5], suggested investigating, in detail, the redox homeostasis

in a disease-transmitting insect The characterization of

A gambiaeTrxR allows the comparison of three different

mechanisms of Trx reduction in the P falciparum malaria

system, i.e that in the parasite, the human host, and the

insect vector

Isolation ofA gambiae TrxR from insect cells

From 8 mL of pelleted A gambiae cells, 19 U of A

gamb-iaeTrxR activity was extracted Approximately 60% TrxR

was recovered from the 2¢,5¢-ADP–Sepharose affinity

column No GR activity was detected either in the dialyzed

crude extract or in any fraction eluted from the column

SDS/PAGE analysis of the TrxR fraction revealed two

major bands representing apparent molecular masses of

62 kDa and 58 kDa (inset Fig 1A) We observed a decrease

in intensity of the heavy band when the cell extract was

ageing As this process could be prevented by repeated

addition of phenylmethanesulfonyl fluoride, it was

conclu-ded that proteolytic cleavage of the 62 kDa protein yielconclu-ded

a product co-migrating with the 58 kDa band

Sequence analysis by Edman degradation

and mass spectral analysis

Edman degradation of the 58 kDa band resulted in the

N-terminal sequence of 17 residues given in Fig 1B; the

62 kDa protein resisted Edman degradation

For further sequence information on A gambiae TrxR,

we conducted mass spectral analyses of the two bands from

the SDS/PAGE gel To achieve this, the proteins were

subjected to tryptic digestion The patterns of high-yield

peptides (Fig 1B) were indistinguishable, which suggests

that the two electrophoretic bands represent splice isoforms

of A gambiae TrxR (see below) Furthermore, sequence

comparison of the peptides with D melanogaster TrxR-1

confirmed that we had indeed isolated a homologue of the

Drosophilaenzyme [5]

Lack of detectable selenium

A particular point of interest was whether TrxR from

Anophelesis a selenoprotein (like its relatives from humans

and other mammals) [30,31] or whether A gambiae TrxR is

a selenium-free Drosophila-type enzyme In mammalian

TrxR, the C-terminal redox centre is formed by a Cys–

selenocysteine (Sec) pair [22,32] No significant selenium –

less than 0.01 mol per mol of A gambiae TrxR subunit

compared with 0.94 mol per mol of human TrxR subunit –

was determined by atomic absorption spectrometry The

absence of a catalytic Sec residue in A gambiae TrxR was

corroborated by the finding that L-dehydroascorbate –

which is a substrate of the selenium-dependent TrxRs of

mammals [33] – was not a substrate at concentrations up to

5 m

Genomic organization of theA gambiae TrxR gene The genome sequence analysis of A gambiae, reported previously [3], enabled us to address the genomic organiza-tion of the gene (Fig 2) A gambiae trxr occurs in three different splicing variants (AJ459821, AJ549084, AJ549085)

as a single-copy gene on chromosome X In contrast to

D melanogaster TrxR-1 – where the coding sequence is interrupted by three introns – the A gambiae TrxR coding sequences were found to be separated by a single intron corresponding to the proximal intron in D melanogaster TrxR-1 (Fig 2)

The identification of three types of expressed sequence tags (ESTs), varying in the sequence of the first exon only, suggests that three alternative transcription start sites are operative (exons 1–3 of A gambiae trxr in Fig 2) Exon 1 is

Fig 1 Characterization of authentic Anopheles gambiae thioredoxin reductase-1 (TrxR-1) by physicochemical analyses (A) The absorption spectra of 6.6 l M TrxR-1 in the oxidized form E ox (dashed curve) and

in the four-electron reduced form (EH 4 ) (solid curve), which was obtained by adding 33 l M NADPH to the E ox sample In the EH 4

sample, the absorption at wavelengths below 400 nm is largely a result

of excess NADPH The inset shows A gambiae TrxR species in a silver-stained gel after SDS/PAGE In lane 1, the two variants of

A gambiae TrxR isolated from cultured insect cells can be distin-guished; lane 2 shows recombinant A gambiae TrxR-1, and the outer lane marker proteins (B) The results of peptide analyses The DNA-deduced sequence of A gambiae TrxR-1 is shown in standard script Tryptic peptides of authentic enzyme that were identified by MS are underlined and marked in bold These peptides were found in both protein bands shown on lane 1 in the inset The N-terminal sequence (17 residues in bold italics) was identified by Edman degradation of the protein isolated from the major band of the SDS/PAGE gel The minor band of 62 kDa resisted Edman degradation.

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represented in the NCBI database by 14 ESTs that overlap

with exon 4 Joining of exon 1 (encoding the five amino

acids MAPLN) with exon 4 (encoding QENYEY and

further 491 amino acids), leads to a protein of 502 residues

which is the orthologue of D melanogaster TrxR-1 (Figs 1B

and 3) This protein is introduced here as A gambiae TrxR-1

(CAD30858)

Two alternative 5¢ exons – exon 2 and exon 3 – were

identified, each represented by a single EST The N-terminal

segment contributed by exon 2 (MATAVLARPARS

LINVVQCVRLIRTQATVMFA) shows the properties of

a mitochondrial targeting sequence containing the predicted

cleavage site between IRT and QAT The last four amino

acids (VMFA) are not encoded by EST BM603316, but the

correct overlap with exon 4 was proven by sequencing a

PCR product amplified with an oligonucleotide pair specific

for exon 1 and exon 4 (data not shown) The deduced

N-terminal sequence of the putative mitochondrial enzyme

is thus QATVMFA|KENEY, the change from Q to K in

position 5 resulting from splicing

Exon 3 occurs in EST BM583435, which extends into

exon 4 The resulting N-terminal sequence – MAAATAAE|

QENYEY – probably represents a second cytosolic species

of TrxR

As judged by the number of EST sequences and by Edman degradation of enzyme isolated from insect cells,

we can state that A gambiae TrxR-1 is the major isoform

in vivo Similarly to the fruit fly, no GR-like sequence could

be identified in the mosquito genome [34] This is consistent with the absence of detectable genuine GR activity in Anophelescell extracts

Cloning and characterization ofA gambiae TrxR-1 The A gambiae trxr-1 gene was cloned and recombinantly expressed in E coli In SDS/PAGE, this protein co-migrates with wild-type A gambiae TrxR-1 – isolated from cultured Anophelescells or whole insects – at a position representing

a molecular mass of 58 kDa (Fig 1A) The discrepancy between this value and the molecular mass of 54.5 kDa deduced from the amino acid sequence has also been observed in other disulfide reductases; they all show a subunit molecular mass (deduced by SDS/PAGE) that is overestimated by 7–10% The molecular basis for this electrophoretic behaviour is unknown [35]

The identity between authentic and recombinant

A gambiae Trx-1 is supported by the concurrence of deduced and experimentally determined amino acid sequences (Fig 1B) Thus, TrxR-1 from A gambiae con-tains 502 amino acids per subunit; the calculated molecular mass is 54.5 kDa per subunit for the apoprotein and

2· 55.3 kDa for the FAD-containing homodimeric holo-enzyme The protein shares 69% sequence identity with its orthologue from D melanogaster (Fig 3) Both insect enzymes have sequence elements that are typical for large TrxRs, including the flavin-near redox-active Cys–Val– Asn–Val–Gly–Cys motif, as well as a C-terminally located redox centre (Fig 3) In the sequence of A gambiae TrxR-1 and D melanogaster TrxR-1 a sequentially adjacent cys-teine pair (Cys500¢ and Cys501¢) is present Thus, the two insect Trxs known, to date, are typical members of the large TrxR enzyme class characterized by an additional redox centre However, in contrast to mammalian TrxRs or TrxR from P falciparum, this part of the active site is structurally distinct in the insect enzymes Indeed a redox centre formed

by two sequential Cys residues is highly unusual [24]

A gambiae TrxR-1 as a Trx-reducing enzyme TrxR-1 was tested with Trxs from A gambiae (A gambiae Trx-1), D melanogaster (D melanogaster Trx-2), and

P falciparum(P falciparum Trx-1) as oxidizing substrates (Table 1) The catalytic efficiency of A gambiae TrxR-1 is

Fig 2 Genomic organization of the Anopheles gambiae thioredoxin

reductase-1 (TrxR-1) gene in com parison with the Drosophila

melano-gaster TrxR gene In both insects the trxr locus is located on

chro-mosome X Numbered boxes represent exons within the gene Coding

regions are shown in black, untranslated sequences are shaded in grey.

Scale bar divisions are in kilobases A gambiae trxr occurs in three

possible splice isoforms (AJ459821, AJ549084, AJ549085) that differ in

the first exon (exon 1, 2 or 3) joined to exon 4 The orthologous

Drosophila locus, shown below, is similarly organized, except that the

sequence corresponding to exon 4 of A gambiae trxr is interrupted by

two short introns, which results in exons 4–6 Exons 1, 2 or 3 joined to

exons 4–6 yield transcripts RA, RB and

CG2151-RC, respectively In both insect species, exon 1 encodes the N-terminal

sequence of an abundant cytosolic, exon 2 of a mitochondrial, and

exon 3 of a minor cytosolic TrxR form.

Table 1 Kinetic parameters of Anopheles gambiae thioredoxin reductase-1 (TrxR-1) with different thioredoxins and 5,5¢-dithiobis(2-nitrobenzoic acid) (DTNB) All values were determined in 100 m M potassium phosphate buffer, 2 m M EDTA, pH 7.4 As expected, A gambiae Trx-1 was the best substrate of A gambiae TrxR-1, but the k cat /K m value was only marginally better than for thioredoxin-2 (Trx-2) from Drosophila melanogaster Plasmodium falciparum thioredoxin-1 (Trx-1) showed the highest k cat value, but the K m was significantly lower than for the insect thioredoxins DTNB was included as an artificial disulfide substrate which is reduced by most high molecular mass thioredoxin reductases.

Substrate K m (l M ) V max (UÆmg)1) k cat (s)1) k cat /K m ( M )1 Æs)1)

A gambiae Trx-1 8.5 ± 1.5 16.9 ± 1.6 15.4 ± 1.5 1.81 · 10 6

D melanogaster Trx-2 9.0 ± 1.0 15.7 ± 1.3 14.3 ± 1.2 1.58 · 10 6

P falciparum Trx-1 33 ± 5 17.2 ± 1.2 15.7 ± 1.1 0.48 · 10 6

DTNB 700 ± 200 6.0 ± 0.7 5.5 ± 0.6 7.9 · 10 3

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comparable with the value previously determined for

D melanogasterTrxR-1 [5,25] Expectedly, with a Kmvalue

of 8.5 lMand a kcatof 15.4 s)1, A gambiae Trx-1 is the best

substrate of the enzyme but D melanogaster Trx-2, i.e the

orthologue of A gambiae Trx-1 in Drosophila, is an almost

equally good substrate The Km value for the reducing

substrate NADPH was found to be 5.0 lM

By comparison with other TrxRs [21,24,36], we can

delineate the pathways of electrons in A gambiae TrxR-1

during catalysis (Figs 3 and 4) The reducing equivalents

flow from the nicotinamide of NADPH via the flavin and

the pair Cys57/Cys62 to the redox centre Cys500’/Cys501’

of the other subunit, and hence to the disulfide of the

substrate Trx The thiolate of Cys62 in A gambiae

TrxR-1 forms a charge transfer complex with the

reoxidized flavin during catalysis [20,24,37] This charge

transfer gives rise to the absorption band at  530 nm (Fig 1A) and thus to the orange/red colour of stable catalytic intermediates that contain both oxidized flavin and Cys62 as a thiolate Unexpectedly, freshly prepared

A gambiae TrxR was found to be orange/red, which indicated the presence of reduced Cys62 Auto-oxidation

of the A gambiae TrxR-1 preparations was very slow and proceeded over a range of hours to days, finally producing the typical yellow colour of oxidized enzyme Eox (Fig 1A) The freshly isolated enzyme also resisted oxidation by its native substrate, A gambiae Trx In contrast, most disulfide reductases, when present in reduced forms, can be easily oxidized by their native substrates [20] Consequently, we conducted redox titration experiments on A gambiae TrxR-1, starting out with the oxidized form, Eox, and monitoring the absorbance

Fig 3 Multiple sequence alignment ( CLUSTAL W ) of high molecular mass thioredoxin reductases (TrxR) The search was conducted (NCBI accession numbers in parentheses) with the enzymes from Anopheles gambiae (AgTrxR-1, CAD30858), Drosophila melanogaster (DmTrxR-1, AAG25639), humans (hTrxR, AAB35418), and from the malaria parasite Plasmodium falciparum (PfTrxR-1, CAA60574) The enzyme of Anopheles shares 69%, 52%, and 45% sequence identity with the TrxRs of D melanogaster, humans, and P falciparum, respectively The sequences of the redox-active centres are shaded in grey; U (residue 498) in the human enzyme represents selenocysteine (Sec).

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at 530 nm As shown in Fig 1A, the absorption coefficient

at 530 nm was 0.4 mM )1Æcm)1for the oxidized enzyme, Eox,

1.6 mM )1Æcm)1after addition of one equivalent NADPH,

leading to the two-electron reduced enzyme species EH2,

and 3.0 mM )1Æcm)1 for the enzyme reduced with two or

more equivalents NADPH, giving rise to the four-electron

reduced enzyme species EH4 After reoxidation with 100 lM

A gambiaeTrx-1, the e-value fell to 1.6 mM )1Æcm)1,

indi-cative of a two-electron reduced enzyme species (EH2) In

contrast, reoxidation of EH4 with 125 lM potassium

ferricyanide, as described previously [16,24], led to the Eox

species with an e-value of 0.4 mM )1Æcm)1at 530 nm

These data confirm that the native substrate does not

reoxidize the enzyme to Eoxbut only to the EH2state where

the redox-active Cys residues 57, 62, 500’, and 501’ are

present partly as thiols so that the thiolate of Cys62 can still

form a charge transfer complex with flavin For catalysis,

this implies that the very first catalytic cycle is primed by two

NADPH molecules, which results in the four-electron

reduced state Oxidation with TrxS2then leads to the

two-electron reduced state, where the two disulfide bridges

are partially reduced, i.e.: priming reaction, Eox+

2NADPH + 2H+fi EH4+ 2NADP+; catalytic cycle,

EH4+ TrxS2fi EH2+ Trx(SH)2; EH2+ NADPH +

H+fi EH4+ NADP+ TrxS2+ NADPH + H+ fi

Trx(SH)2+ NADP+ This balance reaction of the

cata-lytic cycle, of course, represents the net reaction catalyzed by

TrxR

Discussion

The isolation and characterization of A gambiae TrxR contributes to the understanding of the redox metabolism in Diptera The principles of redox homeostasis that were tentatively postulated for the fruit fly can be extended to a disease-transmitting insect In short, a GR is absent, although GSH is a key compound of the redox networks also in insects [38] The nonenzymatic reduction of GSSG

by reduced Trx is probably a major pathway for GSH reduction in these organisms [5] Thus, TrxR indirectly substitutes for the function of GR As described previously,

A gambiaeTrx-1 is a highly expressed protein in vivo [25] The efficiency of GSSG reduction by A gambiae Trx-1 is similar to its orthologue (Trx-2) in D melanogaster and probably sufficient to maintain physiological needs

In the Anopheles mosquito, one TrxR gene is present which occurs in three splice variants Alternative use of first exons was previously reported for mammalian and Droso-phila TrxRgenes [39] In D melanogaster, three alternative transcripts have been identified: CG2151-RA is the ortho-logue of A gambiae TrxR-1; CG2151-RB corresponds to a mitochondrial TrxR form; and CG2151-RC encodes a second cytosolic TrxR Transcripts coding for the latter form are rare, as only two ESTs corresponding to

CG2151-RC have been identified (compared with more than 80 CG2151-RA ESTs) Thus, the trxr loci in Anopheles and

in Drosophila are structurally organized in a similar way: there are three alternative first exons, coding probably for a major cytosolic, a mitochondrial, and a minor cytosolic form (Fig 2)

Unlike in A gambiae, a second TrxR gene, trxr-2, was identified in the genome of D melanogaster However, a

D melanogasterTrxR-1 null mutant leads to death, at the latest during the second larval instar [40], and both cytosolic and mitochondrial TrxR-1 forms have been shown to be necessary for survival [41] Thus, the putative activity of TrxR-2 is not sufficient to compensate for the lackof either the cytosolic or the mitochondrial TrxR-1

The A gambiae TrxR preparation from insect cells results in two enzyme species that can be distinguished by SDS/PAGE (Fig 1) The predominant band represents the cytosolic variant A gambiae TrxR-1, and the 62 kDa band

is possibly the mitochondrial precursor variant This assumption is supported by the size of the protein and by the observation that it is stabilized by protease inhibitors

A gambiae TrxR-1 shares 69% sequence identity with

D melanogaster TrxR-1, including the important redox-active Cys–Cys motif on the C-terminal extension (Figs 3 and 4) For the Drosophila enzyme it was shown that both cysteines are essential for the interaction with the natural substrate Trx [5,24] In the case of rat TrxR, which is a selenoprotein with a Cys–Sec–sequence instead, the Secfi Cys exchange results in a mutant with less than 1% catalytic activity when compared with the wild-type enzyme [30,42] The main difference between the insect enzymes and the TrxR from rat concerns the amino acid residues adjacent to the cysteines In mammalian TrxRs, including the human orthologue, we find a Gly– Cys–Cys–Gly sequence, whereas in A gambiae TrxR-1, it is Thr–Cys–Cys–Ser and in D melanogaster TrxR-1 it is Ser– Cys–Cys–Ser There is evidence that the hydroxyl functions

Fig 4 Sketch of homodimeric Anopheles gambiae in the four-electron

reduced (EH 4 ) form The dimer interface is shown as a diagonal line

with a blackcircle at the centre This circle represents the molecular

two-fold symmetry axis The sketch shows the EH 4 form where all

four redox-active cysteines are present in the reduced form The

thiolate 62 forms a charge transfer complex with oxidized flavin,

and Cys501¢ is ready to attackthe disulfide bond of the substrate

thioredoxin disulfide By analogy with other disulfide reductases,

the most probable scenario leading from oxidized enzyme (E ox ) to

EH 4 is as follows When NADPH binds to one subunit (upper

right), its reducing equivalents flow via the flavin to the disulfide

Cys62/Cys57 The resulting dithiol is reoxidized by exchange with

the disulfide bridge between residues 500¢ and 501¢ of the other

subunit Subsequently, binding and oxidation of a second NADPH

molecule leads to re-reduction of the disulfide between Cys57 and

Cys62.

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of the flanking amino acid residues are crucial for catalysis

(H S Gromer et al., unpublished data) It is interesting to

note that despite the evolutionary distance of 250 million

years [34] between the fruit fly and the mosquito, not only

the basic principles of redox metabolism and the genomic

organization of the TrxR gene, but also the mechanistic

peculiarities of these orthologous enzymes, have remained

highly conserved

With a kcatof 15–22 s)1for Trx [5,24], the insect TrxRs

exhibit a somewhat lower turnover number than their

mammalian relatives ( 35–45 s)1) [30,43], but they have

the advantage of being independent of the rare trace element

selenium This evolutionary adaptation is plausible because

TrxR is apparently the mainstay enzyme of the antioxidant

metabolism in insects, whereas mammals have a second,

GR-based system

Redox processes also represent an interesting aspect of

parasite–host interaction For human malaria it is well

known that disturbance of the antioxidative metabolism

results in an inhibition of parasitic growth in erythrocytes

The most prominent example is

glucose-6-phosphate-dehy-drogenase (Glc6PDH) deficiency, an inherited disease also

known as favism [44,45] A major effect of Glc6PDH

deficiency is an impaired NADPH production, which

affects the ability of the erythrocyte to resist oxidative

stress The effects of Glc6PDH deficiency can be imitated by

GR-inhibitors such as carmustine [46] or isoalloxazines [47]

The current interpretation of Glc6PDH deficiency, as a

condition protecting from severe malaria, is based on the

observation that the anion channel protein of the

erythro-cyte membrane undergoes oxidative changes when

ring-stage parasites are present in the red blood cell This

oxidized band-3 protein is recognized by a specific antibody

that initiates immunologic processes to eliminate the

parasitized cell [48,49]

With respect to the insect vector A gambiae, it has also

been shown that nitrosative and oxidative stress imposed

by NO and peroxynitrite play a dominating role in the

host’s defence against the parasite [50,51] The insect cells,

in turn, have to protect themselves against these reactive

agents When Anopheles cells are challenged by oxidative

stress, transcription of numerous genes that are associated

with the Trx system are induced, prominently among

them the TrxR gene [52] Interestingly, a similar response

occurs after exposing the cells to bacterial peptidoglycan

Trx system-related genes are also transcribed in the

salivary glands of A gambiae It is assumed that the

corresponding proteins are especially important for

pro-tecting the glands from heme-driven free radical attack

[53] Thus, redox processes play a major role in host–

parasite interactions, not only in human blood but also

inside the insect vector

The differences between the enzyme systems involved in

antioxidative metabolism offer an interesting novel target

for the development of insect-specific TrxR inhibitors The

potential of TrxR as a target for novel insecticides is

supported by the fact that TrxR-1 knockouts in Drosophila

are lethal in early embryonic stages [40] Based on the

genomic data and comparative genome analyses, it can be

reasonably assumed that this is also true for Anopheles The

development of novel insecticides is an important approach

in the fight against malaria which is becoming more and

more complicated, not least as a result of the occurrence of insecticide resistance [54] In this context it should be noted that inhibitors of the Trx system have toxic effects themselves but, in addition, they sensitize organisms for other toxic agents [13,55,56] Consequently, inhibitors of

A gambiae TrxR-1 are expected to protect other insecti-cides from the development of resistance

Acknowledgements

We are indebted to Dr Stefan M Kanzokfor the early studies on the Anopheles thioredoxin system and for his help in database searches Our workwas supported by the Deutsche Forschungsgemeinschaft (Grants B2 and C1 of SFB 544 Control of Tropical Infectious Diseases to R.H.S and H.M.M., respectively, as well as grant GR2028/1-1 to S.G.) and by the Fonds der Chemischen Industrie (Grant 161576) to R.H.S.

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