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Tiêu đề Inorganic pyrophosphatase in the roundworm Ascaris and its role in the development and molting process of the larval stage parasites
Tác giả M. Khyrul Islam, Takeharu Miyoshi, Harue Kasuga-Aoki, Takashi Isobe, Takeshi Arakawa, Yasunobu Matsumoto, Naotoshi Tsuji
Trường học National Institute of Animal Health, National Agricultural Research Organization
Chuyên ngành Parasitology
Thể loại Research article
Năm xuất bản 2003
Thành phố Tsukuba
Định dạng
Số trang 13
Dung lượng 638,24 KB

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Amino acid sequence alignment and phylogenetic analysis indicates that the gene encodes a functional Family I soluble PPase containing features identical to those of prokaryotic, plant a

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Inorganic pyrophosphatase in the roundworm Ascaris

and its role in the development and molting process

of the larval stage parasites

M Khyrul Islam1, Takeharu Miyoshi1, Harue Kasuga-Aoki1, Takashi Isobe1, Takeshi Arakawa2,

Yasunobu Matsumoto3and Naotoshi Tsuji1

1

Laboratory of Parasitic Diseases, National Institute of Animal Health, National Agricultural Research Organization, 3-1-5, Kannondai, Tsukuba, Ibaraki, Japan;2Division of Molecular Microbiology, Center of Molecular Biosciences, University of the Ryukyus, Senbaru, Okinawa, Japan;3Laboratory of Global Animal Resource Science, Graduate School of Agricultural and Life Sciences, University of Tokyo, Yayoi, Bunkyo, Tokyo, Japan

Inorganic pyrophosphatase (PPase) is an important

enzyme that catalyzes the hydrolysis of inorganic

pyro-phosphate (PPi) into ortho-phosphate (Pi) We report

here the molecular cloning and characterization of a gene

encoding the soluble PPase of the roundworm Ascaris

suum The predicted A suum PPase consists of 360 amino

acids with a molecular mass of 40.6 kDa and a pI of 7.1

Amino acid sequence alignment and phylogenetic analysis

indicates that the gene encodes a functional Family I

soluble PPase containing features identical to those of

prokaryotic, plant and animal/fungal soluble PPases The

Escherichia coli-expressed recombinant enzyme has a

spe-cific activity of 937 lmol PiÆmin)1Æmg)1 protein

corres-ponding to a kcatvalue of 638 s)1at 55C Its activity was

strongly dependent on Mg2+and was inhibited by Ca2+

Native PPases were expressed in all developmental stages of

A suum A homolog was also detected in the most closely related human and dog roundworms A lumbricoides and Toxocara canis, respectively The enzyme was intensely localized in the body wall, gut epithelium, ovary and uterus

of adult female worms We observed that native PPase activity together with development and molting in vitro of

A suum L3 to L4 were efficiently inhibited in a dose-dependent manner by imidodiphosphate and sodium fluoride, which are potent inhibitor of both soluble- and membrane-bound H+-PPases The studies provide evi-dence that the PPases are novel enzymes in the roundworm Ascaris, and may have crucial role in the development and molting process

Keywords: roundworm; inorganic pyrophosphatase; sodium fluoride; imidodiphosphate; molting

Geohelminth parasites are among the commonest and

widespread of human infections, particularly in the regions

where public health hygiene and nutritional status are

poorly maintained The most prevalent geohelminth is

Ascaris lumbricoides (originally described by Linnaeus in

1758), which colonizes the small intestine of children, and is

estimated to infect a quarter of the world’s population [1]

Ascaris suum(originally described by Goeze in 1782) of pigs

is a very closely related species to A lumbricoides, which can

develop in human hosts, indicating its zoonotic significance [2,3] Childhood infections with Ascaris worms are reported

to be associated with stunting growth, malabsorption, deficiencies of macro- and micro-nutrients and damage of the small intestinal mucosa [4,5] In addition, concurrent Ascarisinfection may have potential immunomodulatory effects on the immune response to other infections [6,7] It

is therefore of considerable interest to investigate the biochemical aspects of Ascaris worms to identify potential drug targets and vaccine candidates

Inorganic pyrophosphatases (PPases), which catalyze the hydrolysis of inorganic pyrophosphate (PPi) into inorganic ortho-phosphate (Pi), are widely distributed among living cells The enzymes play an important role in energy metabolism, providing a thermodynamic pull for many biosynthetic reactions [8], and have been shown to be essential to life [9–11] There are two major categories of PPases, the soluble PPases and the membrane-bound H+ -translocating PPases (H+-PPase) Two families of soluble PPases have been recognized to date, Family I includes most

of the currently known soluble PPases [12], and Family II comprises recently discovered Bacillus subtilis PPase as well

as PPases of four other putative members, two streptococcal and two archeal [13,14] These two families do not show any sequence similarity to each other Family I soluble PPases have been further divided into three subfamilies,

Correspondence to N Tsuji, Laboratory of Parasitic Diseases,

National Institute of Animal Health, National Agricultural Research

Organization, 3-1-5 Kannondai, Tsukuba, Ibaraki 305-0856, Japan.

Fax: + 81 29 8387749, Tel.: + 81 29 8387749,

E-mail: tsujin@affrc.go.jp

Abbreviations: PPase, inorganic pyrophosphatase; H+-PPase,

proton-translocating pyrophosphatase; AsPPase, Ascaris suum inorganic

pyrophosphatase; rAsPPase, recombinant A suum inorganic

pyro-phosphatase; L3, third-stage infective larvae; L4, fourth-stage larvae;

ES, excretory and secretory; IDP, imidodiphosphate.

Enzyme: Soluble inorganic pyrophosphatase (EC 3.6.1.1).

Note: The nucleotide sequences reported in this paper has been

submitted to the DDBJ/EMBL/GenBank with accession number

AB091401.

(Received 4 March 2003, revised 7 May 2003, accepted 9 May 2003)

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prokaryotic, plant and animal/fungal PPases Among the

subfamilies, plant PPases bear a closer similarity to

prok-aryotic than to animal/fungal PPases [12] The H+-PPases,

which appear to work as a reversible H+-pump, are much

larger and do not have any sequence similarity to either of

the two families of soluble PPases [15–17] All known soluble

PPases are homologous proteins, whose active site residues

are highly conserved evolutionarily [18] Site-directed

mut-agenesis and high-resolution X-ray crystallography studies

on PPases from E coli and Saccharomyces cerevisiae have

implicated 17 amino acid residues as being important for

enzyme activity [19,20] However, depending on the choice

of alignment parameters, 14–17 of the putative active site

residues described by Terzyan et al [21] are conserved in

sequence alignments of E coli and yeast PPases [18] Recent

studies have demonstrated that only 17 residues are

conserved in all currently known Family I soluble PPases,

of which 13 are functionally important active site residues

[12,13] PPases are strongly divalent metal ion-dependent,

with Mg2+ conferring the highest PPi hydrolysis activity

[22] Mg2+has several roles: it activates the enzyme and, as a

Mg2+-PPi complex, forms a true substrate for soluble

PPases; Mg2+also stabilizes the enzyme Ca2+is reported

to be a potent inhibitor of soluble PPases [23]

While PPases from diversified sources have been

des-cribed in some detail, no PPase has ever been studied in any

metazoan helminth parasite including the roundworm

Ascaris To address this, we describe here the cloning,

sequencing and heterologous expression in E coli of a gene

encoding PPase of A suum The amino acid sequence of

A suumPPase (AsPPase) indicates that it is an authentic

member of the Family I soluble PPases We also provide

information concerning the kinetics and properties of the

enzyme More strikingly, we show a novel role of the PPase

enzyme in the development and molting process of A suum

larvae in vitro

Materials and methods

Parasites

Adult A suum were obtained from infected pigs at a

slaughterhouse in Shimotsuma, Japan Adult A

lumbric-oides and T canis were obtained from patients after

treatment with piperazine in Bac Gian, Vietnam and, from

an infected dog in Miyazaki, Japan, respectively

Unem-bryonated and emUnem-bryonated eggs were obtained essentially

as described elsewhere [24] Third-stage infective larvae (L3)

from embryonated eggs and lung-stage L3 were obtained as

described previously [25,26] Excretory and secretory (ES)

products from L3, lung-stage L3 and adult worms were

collected as described previously [27] Animal studies were

performed in accordance with the approval of the National

Institute of Animal Health Animal Care and Use

Commit-tee (Approval no 23)

RNA was isolated from embryonated eggs using an

RNA isolation kit (Clontech) Poly(A)+ mRNA was

prepared from total RNA using a polytract mRNA

isolation kit (Clontech) and first-strand cDNA synthesis

was performed using a cDNA synthesis kit and an oligo

(dT)15 primer from Amersham Pharmacia Biotech An

A suumadult female worm cDNA expression library was

constructed in UniZap XR vector (Stratagene) according to the manufacturer’s instructions as previously described [28] Protein concentrations of NaCl/Pi-soluble parasite antigens and ES products were measured using Micro BCA protein assay reagent (Pierce)

Immunoscreening of a cDNA expression library

An adult female worm cDNA expression library was immunoscreened with rabbit antibodies raised against

A suumembryonated egg trickle inoculations Phages were plated onto a lawn of E coli XL-1 Blue at a density of

50 000 phage per dish and grown at 37C for 4 h When plaques were visible, isopropyl thio-b-D -galactoside-impreg-nated filters were placed on the plates for 3 h to obtain a plaque lift After blocking in Tris/HCl, pH 8.0, with 0.05% Tween 20, the filters were incubated in rabbit immune sera overnight at 4C Antibody reactivity with recombinant proteins was revealed by incubation of the filters with alkaline phosphate-conjugated goat anti-rabbit IgG (ICN) for 1 h and developed with 5-bromo-4-chloro-3-indolyl-phosphate/nitroblue tetrazolium (Gibco/BRL) Clones that were reactive with the antibody were plaque-purified by repeated cycles of immune selection Plaque-purified clones were converted using ExAssistTMhelper phages and SOLR

E coli (Stratagene) according to the in vivo excision protocol described in the Stratagene ZAP-cDNA Synthesis Kit (Stratagene) The nucleotide sequences of the cDNAs were determined by the Sanger dideoxy chain termination method using a PRISMTMReady Dye Terminator Cycle Sequencing Kit (PerkinElmer) DNA samples were ana-lyzed using an automated sequencer (373A DNA sequencer, Applied Biosystems).BLASTX(NCBI, National Institute of Health) searches were performed to obtain cDNA clones coding low similarity against mammalian proteins stored at the current database TheGENETYX-WINTMDNA Sequence Analysis Software System (Software Inc) and the BLAST network server of the National Center for Biotechnology Information (NCBI) were used to analyze the nucleotides and deduce the amino acid sequences in determining similarities with previously reported sequences in GenBank

A primary sequence motif was identified using thePROSITE network server at EMBL Analysis of the signal sequence was performed using SIGNALP v1.1 at the Center for Biological Sequence Analysis (http://www.cbs.dtu.dk/ services/SignalP/index.html) Sequences were aligned by the program CLUSTALW1.8 (http://www.ebi.ac.uk/clustalw/) with theBLOSUMamino acid substitution matrix using gap penalties of 10.0 and 0.05 for gap opening and extension, respectively Phylogenetic trees were generated from homologies of the PPase amino acid sequences by the neighbor-joining method and the confidence of the branch-ing order was verified by makbranch-ing 1000 bootstrap replicates using the program CLUSTALW 1.8 The tree was viewed and converted to graphic format with TREEVIEW (http:// taxonomy.zoology.gla.ac.uk/rod/treeview.html)

Expression and purification of recombinant AsPPase proteins

A full length cDNA (lacking signal peptides) was amplified

by PCR as previously described [29] A sense primer

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AATCT-3¢) containing an XhoI (Promega) site upstream of

the start codon and an antisense primer (5¢-CAGCCAA

GCTTCTCACTCTTTGATGAAATGCATCT-3¢)

con-taining a HindIII (Promega) site just downstream of amino

acid residue were used The PCR fragments digested with

XhoI and HindIII were ligated into plasmid expression

vector pTrcHisBTM (Invitrogen), which had also been

digested with the same enzymes according to the

manufac-turer’s instructions The resultant plasmid was transformed

into E coli strain TOP10F¢ (Invitrogen) The transformed

cells were grown to a D600 at 37C in SOB medium

supplemented with 50 lgÆmL)1ampicillin To induce

pro-tein expression, isopropyl thio-b-D-galactoside was then

added to a final concentration of 1 mMand the culture was

grown for an additional 4 h at 37C The E coli cells were

pelleted and resuspended in lysis buffer [50 mMNaH2PO4

(pH 8.0), 10 mMTris/HCl (pH 8.0), 100 mMNaCl]

Lyso-zyme was added at 100 lgÆmL)1, and the cell suspension

was incubated on ice for 15 min The cell suspension was

disrupted using an ultrasonic processor (VP-5, TAITEC) on

ice The E coli lysate was centrifuged at 26 000 g for 30 min

at 4C The supernatants containing recombinant proteins

of AsPPase were purified using ProBondTMresin

(Invitro-gen) under nondenaturing conditions and subsequently

eluted with a stepwise gradient of imidazole (50–500 mM)

The eluted fractions were concentrated by Centrisart I (cut

off MW 10 000; Sartorius) and then dialyzed extensively at

4C with several successive changes of 20 mM Tris/HCl,

pH 7.5 and a decreasing concentration of NaCl in a

Slide-A-Lyzer Dialysis Cassette (Pierce) Fractions were collected

and the presence and purity of recombinant protein was

detected by 10% SDS/PAGE [30] and immunoblot [31]

using anti-T7 tag Ig (Invitrogen) Protein concentrations

were measured with the Micro BCA protein assay reagent

(Pierce)

Production of mouse polyclonal antibodies

BALB/c mice were immunized first with a subcutaneous

injection of 50 lg of recombinant AsPPase (rAsPPase)

emulsified with TiterMax GoldTM (CytRx), followed by

another injection 2 weeks later in the same adjuvant The

mice were bled 2 weeks after the second injection The

antisera from the mice were mixed and stored at)20 C

until used Anti-(mouse rAsPPase) IgG from immune sera

and mouse preimmune IgG were affinity purified using

UltraLinkTMimmobilized protein G according to

manu-facturer’s instructions (Pierce) and used for evaluating the

native AsPPase-neutralizing activity

Two-dimensional electrophoresis

Parasite extracts were treated with an equal volume of urea

mixture consisting of 9M urea, 4% Nonidet P-40, 0.8%

ampholine (pH 3.5–10; Pharmacia) and 2%

2-mercapto-ethanol, and then subjected to 2D PAGE Nonequilibrium

pH gradient electrophoresis was performed [32] in the first

dimension using a rectangular gel electrophoresis apparatus

(AE-6050 A; ATTO) After electrophoresis at 400 V for

2 h, the gels were incubated in the equilibration buffer

for 10 min on a shaker Electrophoresis in the second

dimension was performed on 8% SDS/PAGE gels under reducing conditions The proteins were either stained using

a silver staining kit (Dai-ichi Pure Chemicals) or transferred

to nitrocellulose membranes

Immunoblot analysis Immunoblot analysis was carried out as previously des-cribed [29] Anti-(mouse rAsPPase) serum was used at a dilution of 1: 500 The proteins bound to the secondary antibody were visualized with 5-bromo-4-chloro-3-indolyl-phosphate/nitroblue tetrazolium

Immunohistochemistry Adult females of A suum and A lumbricoides were fixed in 4% paraformaldehyde in 0.1Mphosphate buffer, pH 7.2, overnight and embedded in paraffin Thin transverse sections were made from paraffin-embedded fixed worms The sections on glass slides were deparaffinized and dehydrated using a graded series of alcohol and then rehydrated in NaCl/Pi The slides were blocked for 30 min

in 1% H2O2in NaCl/Picontaining 10% ethanol to inacti-vate endogenous peroxidases For immunolocalization, the slides were blocked in NaCl/Picontaining 10% (v/v) goat serum (Wako) for 30 min at room temperature They were then flooded with anti-(mouse rAsPPase) Ig diluted 1 : 100

in NaCl/Pi/E coli lysate, overnight at 4C Afterwards, the slides were rinsed thoroughly with NaCl/Pi, and the antibody binding was resolved with a peroxidase-labeled anti-mouse IgG and the substrate 3¢,3¢-diaminobenzidine tetrahydrochloride (Sigma FastTM tablets, Sigma) After color development, the slides were dehydrated in a graded series of alcohol and cleared in xylene The slides were then covered with cover slips and observed with a microscope (Axiophot; Carl Zeiss)

Enzyme assay The rAsPPase activity was determined spectrophotometri-cally by measuring the rate of liberation of Pi from PPi using a molybdate-blue based colorimetric assay [33] The recombinant protein was assayed in the standard reaction mixture containing 5 mM Mg2+, 100 mM Tris/HCl (pH 7.5) and 1 mM PPi (Na4P2O7), in a total volume of

200 lL together with the protein solution, at 55C The assay was started by adding 10 lL of diluted rAsPPase solution into the standard reaction mixture The reaction was stopped by adding 1 mL of 200 mM glycine/HCl,

pH 3.0 Then, 125 lL of 1% ammonium molybdate (in

25 mMH2SO4) and 125 lL of 1% ascorbic acid (in 0.05% KHSO4) were added to the mixture, and incubated for

30 min at 37C Protein concentrations and reaction times were chosen in order to obtain the linearity of the reactions

As positive and negative controls, pure yeast-soluble PPase from Sigma (1-1643) and an unrelated A suum 14-kDa recombinant protein (As14; [28]) were used, respectively The concentrations of individual components were varied as indicated for the determination of Mg2+and pH dependent rAsPPase activity The amount of Pi liberated from the hydrolysis of PPi during the course of the reaction was measured in comparison to a standard P sample using a

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spectrophotometer (Model 600, Shimadzu) at an optical

density of 700 nm The specific activity of rAsPPase was

defined as lmol Pireleased min)1Æmg)1of protein

Enzyme kinetic study

The Km(Michaelis constant) and Vmax(maximum velocity)

values were determined by incubating the diluted

recombin-ant proteins in the standard reaction mixture in the presence

of increasing concentrations of PPi(0.05–0.5 mM) at 55C

Data were fit to the appropriate equation using GRAFIT

version 3.09b (Erithacus Software) Kmand Vmaxvalues were

reported with their standard errors derived from the fit

Native AsPPase activity and NaF sensitivity

To investigate the native AsPPase activity during A suum

larval development and molting, the L3 soluble extracts (in

20 mM Tris/HCl, pH 7.5) and the L3 ES products in the

culture fluids (dialyzed against 20 mM Tris/HCl, pH 7.5),

were assayed in the standard reaction mixture as described

above Anti-(mouse rAsPPase) IgG were evaluated for

AsPPase-neutralizing activity Recombinant AsPPase

pro-teins or A suum L3 extracts were preincubated in the

standard reaction mixture containing 5 lgÆmL)1

pre-immune or anti-(mouse rAsPPase) IgG (15 min, 37C)

before adding PPi The sensitivity of the native AsPPase to

inhibition by sodium fluoride (NaF, S-7920; Sigma) was

tested in the present study The L3 extracts were assayed in

the standard reaction mixture for PPi hydrolysis, in the

presence of increasing concentrations of NaF The PPase

activated PPihydrolysis rate was then calculated

Larval molting inhibition assay

To confirm whether the native PPase enzyme is involved in

the molting process, we examined the effects of two PPase

specific inhibitors, imidodiphosphate (IDP, 1-0631; Sigma)

and NaF on development and molting of A suum

lung-stage L3 to fourth-lung-stage larvae (L4) in vitro The lung-lung-stage

L3 were obtained from the lungs of New Zealand white

rabbits 7 days after inoculation with 2.5· 105embryonated

infective eggs of A suum [26] Briefly, the rabbits were killed

by an overdose of ketamine hydrochloride (50 mgÆkg)1

body weight, i.v.) followed immediately by decapitation and

the lungs were removed and minced with a surgical knife

The minced tissue was wrapped in cotton gauze and

suspended in NaCl/Pi containing 100 lgÆmL)1 penicillin/

streptomycin at 37C for 3 h After incubation the tissues

were removed, and the larvae were collected from the

bottom of the tube The recovered L3 were washed several

times with warm NaCl/Picontaining 50 lgÆmL)1penicillin/

streptomycin, and subjected to molting inhibition assay

Briefly, 50–100 L3 in 1 mL of RPMI 1640 medium (Gibco/

BRL), pH 6.8 supplemented with 10% (v/v) fetal bovine

serum (Sigma), 50 lgÆmL)1 penicillin/streptomycin were

cultured in 24-well flat-bottomed tissue culture plates

(Costar) The cultures were incubated at 37 C in a

humidified 5% CO2incubator in the absence (control) and

presence of increasing concentrations of inhibitors for

10 days, and the number of molting larvae was determined

Molting was manifested by shedding of the L3 cuticle

Numbers of molted larvae in a culture well were therefore determined by counting the L3 cuticles shed from the larvae Furthermore, molted larvae (that had already shed their cuticles) exhibited an intense motility compared with unmolted larvae (that had not shed their cuticle) Aliquots

of larvae were removed at different days of postcultures and photographs were taken

Results

Identification of cDNA encodingA suum inorganic pyrophosphatases

A clone designated AdR44 was isolated initially by immu-noscreening an A suum female worm cDNA library with serum obtained from a rabbit immunized with A suum infective eggs AdR44 was selected for further characteriza-tion because of its sequence homology to the inorganic pyrophosphatase family of proteins Sequence analysis showed that AdR44 was 1,375-bp long with an open reading frame (ORF) coding for 360 amino acids The ATG initiation codon is predicted to be at nucleotides 79–81 and

is followed by a region encoding a hydrophobic sequence of

17 amino acids, which may function as a signal peptide The 3¢ untranslated region contained 224 bp and ended with 17-bp poly(A)+tail that began 14-bp down-stream from the sequence AATAAA, which is the eukaryotic consensus polyadenylation signal An entire ORF of the AdR44 cDNA encodes a sequence of 360 amino acids, predicting a

40 600-Da polypeptide with an isoelectric point of 7.1 Removal of the signal peptide resulted in a putative mature protein with molecular weight 38 771 Da Two potential sites for N-glycosylation (residues 50–53, 246–249) were predicted in AsPPase The three conserved aspartates that are involved in the binding of cations in PPases (D-[SGDN]-D-[PE]-[LIVMF]-D-[LIVMGAC]) were found at position 192–197 A search of the protein database conducted using the information obtained from the NCBI revealed that AsPPase shared a high degree of sequence similarity to those

of animal/fungal PPases in Family I soluble PPases Figure 1 shows a comparison of the AsPPase sequence to five other sequences of animal/fungal soluble PPases The deduced amino acid sequence of AsPPase shows 74% similarity (56% identical) to the Drosophila melanogaster PPase sequence, 69% similarity (55% identical) to the sequence of Caenorhabditis elegans, 72% similarity (55% identical) to the sequence of Schizosaccharomyces pombe, 70% similarity (51% identical) to the sequence of Bos taurus and 67% similarity (51% identical) to the sequence of

S cerevisiae Sequence similarities occur throughout the protein but few are at both ends Sequence analysis revealed that all 13 functionally important active site residues (AsPPase numbering: E-125, K-133, E-135, R-155, Y-170, D-192, D-194, D-197, D-224, D-229, K-231, Y-269 and K-270) (Fig 1), which have been reported previously to be evolutionarily well conserved in Family I soluble PPases [12,13,18,34,35], are identical in AsPPase

Phylogenetic analysis of available PPases

We have constructed a phylogenetic tree using Family I soluble PPase sequences by the neighbor-joining method

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and the confidence of the branching order was verified by

making 1000 bootstrap replicates with the CLUSTALW

program (Fig 2) The neighbor-joined trees reveal that

animal and fungal PPases including AsPPase represent a

separate group from plant and prokaryotic PPases

Fur-thermore, within the animal/fungal subgroup, AsPPase is

more closely clustered with PPases from the free-living

model nematode C elegans and the insect D melanogaster

Characterization of recombinant AsPPase

The gene encoding the soluble PPase of A suum was

amplified by PCR with A suum female worm cDNA

AsPPase was then overexpressed in E coli strain TOP10F¢

using pTrcHisBTMvector, to test whether the clone indeed has an inorganic pyrophosphatase activity Recombinant AsPPase was expressed in E coli with a yield of 1 mgÆL)1of bacterial culture The rAsPPase was 99% pure as determined

by SDS/PAGE analysis The observed molecular mass of rAsPPase corresponded well to the calculated mass of the AdR44 cDNA (data not shown) The functional activity of the purified rAsPPase was determined using a PPihydrolysis assay in a standard reaction mixture containing 5 mMMg2+,

100 mMTris/HCl, pH 7.5 and 1 mMPPi The recombinant protein showed a specific activity of 937 lmol PiÆmin)1Æmg)1 protein corresponding to a kcatvalue of 638 s)1that could be abolished by Ca2+or removal of Mg2+ (Table 1) This activity could not be due to copurification of endogenous

E coliPPase as, the rAsPPase contained a His-tag that was used for purification and the recombinant protein was determined to be pure by SDS/PAGE analysis

Expression and immunohistochemical detection

of native AsPPase

We performed 2D immunoblot analysis to identify native AsPPase in adult female A suum Anti-(mouse rAsPPase) serum reacted strongly with a protein having a molecular mass of 39 kDa with a pI of 7.1 (Fig 3A) confirming that it corresponded to the predicted size of the putative mature protein (38.771 kDa) calculated from the AsPPase amino acid sequence except for a signal peptide In addition, a native AsPPase was identified on silver-stained 2D gels on which more than 200 visible protein spots appeared (data not shown) To determine the N-terminal residues, we excised the native AsPPase spots from 2D immunoblotted polyvinylid-ene difluoride membranes and subjected them to analysis by the automatic Edman degradation method The sequence 1-MALAASATIS-10 of native AsPPase was identical to that

of the putative mature protein This confirmed that our clone encoded a soluble PPase of A suum A spot reacting with the anti-mouse rAsPPase was also seen in parasite extracts and

ES products from various developmental stages, including embryonated eggs, L3, lung-stage L3 and adult male and female worms, indicating that native AsPPases were expressed in all lifecycle stages of A suum (data not shown) Interestingly, enzyme homologs were also expressed in the human roundworm A lumbricoides and the dog roundworm

T canis(Fig 3B) Native AsPPases that reacted with mouse polyclonal antibodies against rAsPPase were localized in various structures such as the hypodermis, dorsal and lateral hypodermalchord, in muscle cells, gut epithelium and, in the uterus and ovary of adult female A suum (Fig 4B–D) No labeling was, however, seen in sections probed with mouse preimmune sera (Fig 4A) This study also detected the ubiquitous presence of AsPPase homologs in various organs

of A lumbricoides (data not shown)

Enzymatic properties of recombinant proteins The PPi dependence of the maximum hydrolytic velocity (Vmax) of the recombinant AsPPase protein was shown to

be 849.005 ± 14.635 lmol PiÆmin)1Æmg)1 protein with a

Km (Michaelis Constant) value of 0.117 ± 0.006 mM for PPi from three independent experiments (Fig 5A) The K value is significantly higher than the values of

Fig 1 Sequence alignment of representative members of Family I

sol-uble PPases CLUSTALW alignment of soluble PPases (GenBank

accession numbers are indicated in parentheses): A suum (AB091401),

C elegans (CAA93107), D melanogaster (O77460), Bos taurus

(P37980), S cerevisiae (2781300) and Schizosaccharomyces pombe

(P19117) Identical residues among PPases are marked with asterisks.

The 13 essential, active site residues that are conserved in all Family I

soluble PPase sequences currently available in the GenBank are further

emphasized by bold typeface The signal peptides are underlined.

Dashes indicate gaps inserted to optimize the alignment The

num-bering is for the sequence of A suum (As) PPase As, A suum, Ce,

C elegans, Dm, D melanogaster, Bt, B taurus, Sc, S cerevisiae, Sp,

S pombe.

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0.0009–0.00147 mM from bovine retinal PPase [23],

0.005 mM from rat liver PPase [36] and 0.026 mM from

bovine rod outer segment PPase [37] These discrepancies in

Kmvalues for PPiare not entirely surprising in view of the

many differences in enzyme purity and assay methodology

The rAsPPase was shown to absolutely require Mg2+for

PPihydrolysis The maximum enzyme activity was found

with 5 mM Mg2+using 1 mM PP, which then gradually

declined with increased concentrations of Mg2+(Fig 5B)

A drop in enzyme activity due to increased concentrations

of Mg2+(>5 mMMg2+) has not been investigated in the present study Although excess of Mg2+is known to inhibit the PPases, however, the mechanism is yet unclear Both

E coli and yeast PPases have four (M1–M4) subsites for binding metal ions for catalysis [38,39] It has been urgued that binding of Mg2+ at three subsites is required for

Fig 2 Phylogenetic tree based on alignment of available Family I soluble PPase sequences The sequences shown are those from (GenBank accession numbers are indicated in parentheses): A suum (AB091401), S cerevisiae (2781300), Kluyveromyces lactis (P13998), Pichia pastoris (O13505),

S pombe (P19117), D melanogaster (O77460), C elegans (CAA93107), B taurus (P37980), Homo sapiens, from ([12]), S cerevisiae mitochondria (P28239), Hordeum vulgare (O23979), Zea mays (O48556), Solanum tuberosum (O43187), Arabidopsis thaliana (AAC33503), Oryza sativa (AAC78101), Chlamydia pneumoniae (AAD19056), Chlamydia trachomatis (O84777), Mycoplasma pneumoniae (P75250), Mycoplasma genitalium (P47593), Bacillus stearothermophilus (BAA19837), Synechocystis (PCC6803, P80507), Thermoplasma acidophilum (P37981), Methanobacterium thermoautotrophicum (O26363), Thermococcus litoralis (P77992), Pyrococcus horikoshii (O59570), T thermophilus (P38576), Mycobacterium leprae (O69540), Mycobacterium tuberculosis (CAB08851), Haemophilus influenzae (1170585), Sulfolobus acidocaldarius (P50308), Aquifex aeolicus (O67501), Helicobacter pylori (P56153), Gluconobacter suboxydans (O05545), Bartonella bacilliformis (P51064), Rickettsia prowazekii (CAA15034), Legionella pneumophila (O34955) and E coli (P17288) The bar indicates the numbers of substitutions per site Unrooted neighbor-joining trees were generated from homologies of soluble PPase sequences and the confidence of the branching order was verified by making 1000 bootstrap replicates using the CLUSTALW program The tree was viewed and converted to graphic format with TREEVIEW

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catalysis to proceed, whereas binding at M4 causes

inhibi-tion [38,40]) Omission of Mg2+from the reaction medium

abolished PPase-mediated PPi hydrolysis The enzyme

activity was found to be optimum in the pH range 7.0–8.0

(Fig 5C) The pH profile showed a dramatic drop in

activity at high pH These results are consistent with other

soluble PPases from various sources [35,36,41]

Detection of native AsPPase activity and inhibition

by IDP and NaF of larval development and molting Native enzyme activity in L3 soluble extracts and in

ES products was detected by PPase-activated PPi hydroly-sis The L3 extracts showed an activity of 1.58 ± 0.02 lmol

PiÆmin)1Æmg)1, whereas L3 ES exhibited that of 0.51 ± 0.03 lmol PiÆmin)1Æmg)1protein for PPihydrolysis Anti-(mouse rAsPPase) IgG partially inhibited recombinant AsPPase (6 ng) activity up to 22% in the presence of

5 lgÆmL)1anti-(mouse rAsPPase) IgG relative to AsPPase activity determined in the presence of 5 lgÆmL)1 mouse preimmune IgG (data not shown) Also, native AsPPase activity in L3 extracts was shown to be partially inhibited (25%) by anti-(mouse rAsPPase) IgG (data not shown) indicating that AsPPase is responsible for the hydrolyzing activity of PPiin A suum L3 extracts NaF, an anion, is a potent inhibitor of PPases and was able to inhibit native AsPPase activity at micromolar concentrations in a dose-dependent manner (Fig 6A) This agent is also known to inhibit the H+-PPases from plants [42], trypanosomatids [43,44] and apicomplexan protozoa [45]

The L3 of A suum develop and molt to L4 in the lungs of their vertebrate hosts that can also occur during in vitro cultivation To determine whether this complex process is regulated by PPase enzyme, we examined IDP, a non-hydrolyzable PPi analogue, and NaF for their possible

in vivoability to inhibit/arrest development and the molting process by blocking the PPase activated PPihydrolysis, as the native PPases in L3 extracts were found to be very sensitive to inhibition by NaF (Fig 6A) As IDP interferes with the colorimetric assay, it was, however, not possible

to examine enzyme sensitivity with this compound in the present study In vitro molting inhibition experiments demonstrated that IDP and NaF inhibited molting of

A suumL3 to L4 with varying success, in a dose-dependent manner (Fig 6B,C) Up to 55% molting was inhibited at a maximum concentration of 10 mMIDP without affecting the growth and viability of the L3 In contrast, NaF inhibited 65% molting at 1 mMconcentration However, at higher concentrations (>1 mMNaF) molting inhibition was increased drastically up to 100% with an apparent growth inhibition of the L3 observed on day 5 postculture and onwards A mild larvicidal effect of 10 mM NaF with progressive damage of the body wall and intestine was seen

on day 5 postculture and onwards (data not shown) The molted L3 developed well to L4 in control culture with increased body length and width (data not shown), and changes in the structure of the head and tail (Fig 7A–C) compared with unmolted L3 which achieved little or no development, being inhibited by IDP/NaF (Fig 7D,E) Under light microscopy, it was however, not possible to detect the formation and/or separation of new cuticles of unmolted L3 exposed to inhibitors that might be carried out

by electron microscopy The mean molting percentage in control culture was recorded as, 52.59 ± 4.12

Discussion

Although PPases are distributed widely among living cells, most of the previous studies have focused on microbial and plant enzymes, and very little is known about the enzyme

Fig 3 Identification of A suum native PPase in adult female worm.

(A) Fifty micrograms of female worm extract was separated by 2D

nonequilibrium pH-gradient gel electrophoresis, and the proteins were

then transferred to a nitrocellulose membrane The native AsPPase

bound to the anti-(mouse rAsPPase) serum was found by alignment of

the stained gel and immunoblot membrane (B) Expression of AsPPase

homologs in ascarid roundworms Sixty or 80 mg of protein

equiva-lents of each parasite extract were electrophoresed on a 10% SDS/

PAGE and blotted onto a nitrocellulose membrane The AsPPase

homologs bound to the anti-(mouse rAsPPase) serum were detected by

5-bromo-4-chloro-3-indolylphosphate/nitroblue tetrazolium Lane 1,

A lumbricoides; lane 2, T canis; lane 3, A suum.

Table 1 Recombinant A suum PPase activity Pyrophosphatase

activity was assayed as described in the Materials and methods section;

–, no detectable activity Data represent the mean ± SE from three

independent experiments An unrelated A suum 14-kDa recombinant

protein was used as the negative control (As 14; [28]).

Assay conditions

Activity (lmol P i Æmin)1Æmg protein)1)

A suum PPase +5.0 m M Mg 2+ 937.76 ± 39.76

A suum PPase +0.0 m M Mg 2+ –

A suum PPase +5.0 m M Ca2+ –

Yeast-PPase +5.0 m M Mg 2+

(positive control)

13232.36 ± 183.42 As14 + 5.0 m M Mg2+

(negative control)

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from mammalian tissues In contrast, we do not have any

evidence of PPases from parasitic helminths We report here

the cloning, sequencing and biochemical and functional

characterization of a novel PPase from the important

pathogenic roundworm A suum The deduced amino acid

sequence of AsPPase shows significant similarity with

animal/fungal PPase sequences in Family I soluble PPases

(Fig 1) All members of Family I soluble PPases currently

available in the database have been shown to contain 13

functionally important active site residues that are

evolu-tionarily well conserved, and were found to be identical in

AsPPase Several highly conserved regions, the most

prominent of which is an eight residue long sequence

(224-DEGETDWK-231), are also seen in the AsPPase

sequence It will be interesting to see the significance of this

highly conserved region in AsPPase structure and

function-ing Over 37 Family I soluble PPases have been identified

The prokaryotic PPases are hexamers of  20 kDa and

reported to contain 162–220 amino acid residues per

subunit, while eukaryotic PPases are dimers of 30–36 kDa

with 211–310 residues per subunit [12,13,18] AsPPase, with

360 amino acid residues having a calculated molecular mass

of 40.6 kDa, resembles eukaryotic PPases and thus is the

largest among the Family I soluble PPases stored in the

current protein database This is largely due to a longer

N-terminal region compared with other PPases (Fig 1)

The membrane-bound H+-PPases that are found in plants

[17], certain bacteria [46], and more recently identified from

trypanosomatids [47] and apicomplexan protozoa [48] differ greatly in structure and function from soluble PPases The

H+-PPases are much larger (660–770 amino acid residues per monomer) and do not have any sequence similarity to soluble forms [15,16,49] The AsPPase described here is clearly a soluble PPase and it does not have any sequence similarity to plant/protist H+-PPases Together, these findings suggest that AsPPase is a distinct Family I soluble PPase Phylogenetically, AsPPase is, within the subfamily

of animal/fungal soluble PPases, closer to C elegans and

D melanogaster PPases than to fungal and mamma-lian PPases (Fig 2) Moreover, E coli-expressed purified rAsPPase protein has shown enzymatic activity (937 lmol PiÆmin)1Æmg protein)1) by PPi hydrolysis assay that was found to be closer to those of the highly purified and crystallized E coli (2000 lmol PiÆmin)1Æmg)1 [50]), yeast (655 lmol PiÆmin)1Æmg)1 [51]), rat liver 600–

700 lmol PiÆmin)1Æmg)1 [36]) and bovine retinal PPases (>885 lmol PiÆmin)1Æmg)1[23]) Thus, AsPPase represents the first member of Family I soluble PPase enzymes to be identified from the parasitic helminths

The rAsPPase activity was shown to be strictly Mg2+ -dependent On the contrary, Ca2+inhibited the activity to some degree in the presence of Mg2+(data not shown) These distinctive features have been well demonstrated for Family I soluble PPases [23,52,53] The rAsPPase enzyme, however, requires a higher concentration of Ca2+ for inhibition (data not shown), and this finding is fairly

Fig 4 Immunohistochemical localization of A suum native PPase in adult female worm A suum female worms were fixed in paraformaldehyde, embedded in paraffin, sectioned (7-lm thickness) and exposed to either mouse preimmune serum as a control (A) or mouse anti-rAsPPase serum diluted 1: 100 (B) (C) and (D) (both 25·) are magnified areas of (B) Arrows indicate antibody-labeled regions; cu, cuticle; hd, hypodermis;

hc, hypodermal chord; mu, muscle; gu, gut; ov, ovary; ut, uterus.

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consistent with other animal PPases [36,54] but contrasts with the reports on yeast PPase and on porcine brain and bovine retinal PPases [23,41] that demonstrated much lower concentrations of Ca2+were needed for enzyme inhibition Prior studies have shown that free PPiis a potent inhibitor, and free Mg2+activates the enzyme and binds with PPito form a true substrate, Mg2+PPifor soluble PPases [38,55] Family II PPases are easily distinguishable from Family I PPases in having a preference for Mn2+over Mg2+as the activator, and are not inhibited by Ca2+, rather Ca2+

Fig 5 PP i (A), Mg2+(B) and pH (C) dependence of A suum

recom-binant PPase activity (A) Diluted recomrecom-binant proteins were run in the

standard reaction mixture (as described in Materials and methods) for

PPase assays at 55 C, in the presence of increasing concentrations of

PP i (0.05–0.5 m M ) Data were analyzed using a computer assisted

program ( GRAFIT version 3.09b) The theoretical curve drawn is for

the best fit values of K m ¼ 0.117 ± 0.006 m M , and V max ¼

849.005 ± 14.635 lmolÆmin)1Æmg protein)1 The inset in (A)

repre-sents the linear transformation of the curve (B) Mg2+dependency was

determined as described in (A) in the presence of increasing

concen-trations of Mg 2+ (C) pH dependency of the enzyme was examined as

described above using several buffers with increasing pH values The

buffers used were (100 m M ), sodium acetate (pH 5.0–5.5), Mops

(pH 6.0–6.5), Tris/HCl (pH 7.0–8.5) and glycine/NaOH (9.0–10.5).

Data represent mean ± SEM from three independent experiments.

Fig 6 Inhibition of A suum native PPase activity and A suum L3 molting by IDP and NaF (A) Aliquots of A suum L3 soluble extracts,

17 mg proteinÆmL)1 was run in the standard reaction mixture for PPase assays at 55 C, in the presence of increasing concentrations

of NaF Percentage activity compared to the control in the absence

of NaF (100%) Control activities were 1.58 ± 0.01 lmol

P i Æmin)1Æmg protein)1 for PP i hydrolysis Data represent mean ± SEM from three independent experiments (B) Lung-stage A suum L3 were cultured for molting inhibition assays, in the presence of increasing concentrations of IDP and (C) NaF Molting percentage was determined on day 10 Percentage activities are relative to the control in the absence of inhibitor (100%) Molting percentage of control was 52.59 ± 4.12 Data represent mean ± SEM of triplicates.

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activates the enzymes preincubated with Mn2+ [13,53]

further indicating that AsPPase reported here is an

authen-tic member of Family I soluble PPases

An intense expression of native AsPPase in metabolically

active tissues, such as the body wall, gut epithelium and

reproductive organs, of adult female worms suggests a

critical role of the enzyme in these organs The presence of

AsPPase in embryonated eggs, L3, lung-stage L3, adult

worms and their ES products together with its direct

detection in L3 soluble extracts and in ES products

(1.58 ± 0.02 lmol PiÆmin)1Æmg)1 and 0.51 ± 0.03 lmol

PiÆmin)1Æmg)1protein for PPihydrolysis, for L3 extracts and

ES products, respectively) strongly suggested the important

roles of the PPase enzyme throughout the developmental

cycle of Ascaris parasites The results of neutralization

studies indicate that AsPPase-specific IgG may interfere

with the development and molting process of Ascaris larvae

We showed that the native AsPPases are very sensitive to

inhibition by NaF in the micromolar range (Fig 6A) This

value is much lower than previously reported data for NaF

against H+-PPases from parasitic protozoa [44] These

results prompted us to investigate the role of PPase enzymes

in the development and molting process of A suum larvae

and to test whether this could be targeted by inhibitors We

used IDP, a nonhydrolyzable PPianalogue, and NaF, a well

known inhibitor of Family I and Family II soluble PPases

(NaF competes with the hydroxide ion for binding to Mg2+

in the active site of the enzyme [35,53]), to block enzyme

activity We demonstrated for the first time that NaF is

highly effective in inhibiting the development and molting of

A suumL3 to L4, in a concentration-dependent manner, whereas, IDP has shown only partial inhibitory effect (Fig 6B,C) However, a much higher concentration of NaF (>1 mM) is required to completely block development and molting of L3 as against micromolar concentration is needed for the inhibition of native enzyme in vitro This difference may in part be attributed to the difference between live parasites and their soluble extracts used in the assay system

We observed that during in vitro cultivation, the L3 could not develop and molt to L4 in the presence of inhibitor, even

at the end when the culture had terminated (Fig 7D,E) These observations indicate that PPase enzymes are prob-ably involved in the development and molting process of

A suumL3 to L4 However, the mechanisms of inhibition of this complex process by PPase inhibitors are yet to be elucidated Although aminopeptidase, cysteine protease and hyaluronidase enzymes so far have been reported to be involved in the development and molting process of A suum L3 to L4 in vitro, virtually none had been characterized in relation to the actual mechanism of the molting process in this roundworm [56] The basic structure of the body wall of parasitic roundworms consists of the cuticle, an underlying syncytial or cellular layer called the hypodermis, and the longitudinally oriented somatic musculature The ecdysis of

an old cuticle and deposition of the components of a new cuticle that are synthesized in the hypodermis and are secreted across the hypodermal membrane into the space between it and the old cuticle, occur at each of four molts during the lifecycle of all roundworms [57] The molecular mechanisms regulating this complex process are, however,

Fig 7 In vitro development and molting of A suum lung-stage L3to L4 in the absence of inhibitor (control; A–C) and its presence (D,E) (A) L3 on day 0 culture from control (B) L3 had initiated molting on day 5 postculture from control Arrow indicates an entirely distended L3 cuticle (C) L4 (molted L3) on day 10 postculture from control A cuticle which had shedded from L3 is indicated by an arrow (D) L3 had not initiated molting on day 5 postculture with inhibitors, IDP/NaF (E) L3 had not molted on day 10 postculture with IDP/NaF Photographs were taken using differential interference contrast microscopy.

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