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Furthermore, it was clearly shown that madanin 1 and 2 inhibited conversion of fibrinogen into fibrin by thrombin, thrombin-catalyzed activation of factor V and factor VIII, and thrombin-i

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Identification and characterization of novel salivary thrombin

Shiroh Iwanaga1, Masakazu Okada1, Haruhiko Isawa3, Akihiro Morita2, Masao Yuda2and Yasuo Chinzei2

1

Laboratory of Chemistry and Utilization of Animal Resources, Faculty of Agriculture, Kobe University, Japan;

2

Department of Medical Zoology, School of Medicine, Mie University, Tsu, Japan

3

Laboratory of Physiology and Biochemistry, Department of Medical Entomology, National Institute of Infectious Diseases, Tokyo, Japan

Novel antithrombin molecules were identified from the

ixodidae tick, Haemaphysalis longicornis These molecules,

named madanin 1 and 2, are 7-kDa proteins and show no

significant similarities to any previously identified proteins

Assays using human plasma showed that madanin 1 and 2

dose-dependently prolonged both activated partial

throm-boplastin time and prothrombin time, indicating that they

inhibit both the intrinsic and extrinsic pathways Direct

binding assay by surface plasmon resonance measurement

demonstrated that madanin 1 and 2 specifically interacted

with thrombin Furthermore, it was clearly shown that

madanin 1 and 2 inhibited conversion of fibrinogen into

fibrin by thrombin, thrombin-catalyzed activation of factor V and factor VIII, and thrombin-induced aggregation

of platelets without affecting thrombin amidolytic activity These results suggest that madanin 1 and 2 bind to the anion-binding exosite 1 on the thrombin molecule, but not to the active cleft, and interfere with the association of fibrinogen, factor V, factor VIII and thrombin receptor on platelets with

an anion-binding exosite 1 They appear to be exosite 1-directed competitive inhibitors

Keywords: anticoagulant; Haemaphysalis longicornis; sali-vary gland; thrombin inhibitor; tick

Thrombin has various physiological functions and plays

important roles in hemostasis For example, in the final step

of blood clot formation, thrombin converts soluble

fibri-nogen into fibrin and subsequently triggers cross-linking

between fibrin monomers by activating factor XIII [1] It

also amplifies its own generation by activating

nonenzy-matic cofactors V and VIII as well as factor XI [2,3]

Conversely, it suppresses its own generation by activating

protein C [4], which inactivates factor Va and factor VIIIa

together with protein S [5], when bound to the endothelial

membrane receptor thrombomodulin In addition,

throm-bin induces platelet aggregation via proteolytic activation of

G-protein-coupled protease-activated receptors (PARs)

[6,7] Specific interactions of thrombin with these substrates,

cofactors, and receptors involve not only the catalytic site

and the primary binding pocket, but also secondary

recognition sites, termed anion-binding exosite 1 and 2

Anion-binding exosite 1 interacts with negatively charged

domains on fibrinogen [8], PARs [6,7,9], and

thrombomo-dulin [10,11] Anion-binding exosite 2 interacts with heparin

[12], promoting inhibition of thrombin by antithrombin III

[13] and heparin cofactor II [14] Furthermore, both exosites

are involved in the recognition of factor V and factor VIII

by thrombin [15]

The salivary glands of blood-sucking animals, such as leeches, insects, and ticks, contain various anticoagulants [16] These substances inhibit the host hemostatic response so that the blood-sucking organism can feed smoothly on host blood The best known anticoagulant identified from blood-sucking organisms is hirudin, a highly specific thrombin inhibitor, isolated from the medical leech, Hirudo medicinalis [17] It interacts with two distinct sites on the thrombin molecule: its N-terminal and C-terminal domains bind to the active site and anion-binding exosite 1, respectively [18,19] This binary binding mechanism appears to contribute to its potent inhibitory activity It has also been demonstrated that the peptide alone derived from the C-terminal domain of hirudin is able to inhibit various thrombin functions [20–23] This indicates that anion-binding exosite 1 is essential in interactions between thrombin and its substrates, and that competitive binding to anion-binding exosite 1 is one strategy

of thrombin inhibition

In this paper, we describe two novel anticoagulants identified from the ixodidae tick, H longicornis These molecules exhibit no sequence similarities to any previously known proteins We show that the recombinant anticoagu-lant molecules clearly prolong both activated partial thromboplastin time (APTT) and prothrombin time (PT), and specifically bind to thrombin We further demonstrate that these molecules inhibit the conversion of fibrinogen into fibrin, activation of factor V and factor VIII, and aggregation of platelets by thrombin without inhibiting thrombin amidolytic activity toward a small synthetic substrate These results suggest that these factors are novel exosite 1 competitive inhibitors like the C-terminal peptide

of hirudin

Correspondence to S Iwanaga, Laboratory of Chemistry and

Utilization of Animal Resources, Faculty of Agriculture,

Kobe University, Kobe 657-8501, Japan.

E-mail: iwanaga@ans.kobe-u.ac.jp

Abbreviations: APTT, activated partial thromboplastin time; PT,

prothrombin time; PAR, protease activated receptor; SPR, surface

plasmon resonance; RU, resonance unit.

Enzyme: Thrombin (EC 3.4.21.5).

(Received 13 November 2002, revised 1 March 2003,

accepted 7 March 2003)

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Experimental procedures

Materials

Human thrombin, bovine thrombin, human factor X/Xa,

human factor IXa, and human factor V were purchased from

Enzyme Research Laboratories The following absorption

coefficients (e0.1%,280) and molecular masses were used to

determine protein concentrations: human thrombin, 18.3,

37 kDa; bovine thrombin, 19.5, 37 kDa; human factor X,

11.6, 58.8 kDa; human factor Xa, 11.6, 46 kDa; human

factor IXa, 14.9, 56 kDa; human factor V, 9.6, 330 kDa

Human factor VIII was obtained from American

Diagnos-tica Inc., Greenwich, CT, USA and the concentration

adjusted to 0.25 UÆlL)1 Human fibrinogen was from

Sigma-Aldrich Chromogenic substrates, S-2238 and

S-2222, were obtained from AB Kabi Restriction enzyme

was purchased from Invitrogen All other reagents were

analytical grade and obtained from either Nacalai Tesque,

Kyoto, Japan or Wako Pure Chemical Industry, Osaka,

Japan

Mass sequence analysis of cDNA clones

H longicornissalivary gland mRNA was isolated from 10

pairs of glands of ticks at three distinct feeding stages

(unfed, slow feeding, and rapid feeding) by using a

QuickPrepMicro mRNA Purification Kit (Amersham

Bioscience) Three cDNA libraries were constructed from

each isolated mRNA using SuperScript Plasmid System for

cDNA Synthesis and Plasmid Cloning Kit (Invitrogen) In

total, approximately 2000 cDNA clones were picked

randomly from three libraries, and their partial DNA

sequences were determined using T7 primer and an ABI

PRISM Big Dye Terminator Cycle Sequencing Kit (Applied

Biosystems) Sequence similarity searches of clones were

carried out using theBLASTprogram at the National Center

for Biotechnology Information (NCBI) In addition, signal

peptides of deduced amino-acid sequences were predicted

using the SIGNAL PProgram at the Center for Biological

Sequence Analysis (CBS)

Expression and purification of recombinant proteins

DNA fragments encoding predicted mature regions of

recombinant proteins were amplified by PCR using each

specific primer set Amplified PCR products were subcloned

into the NdeI–HindIII site of pET22b expression vector

(Novergen) After verification of nucleotide sequences of

constructed plasmids, recombinant proteins were expressed

according to the manufacturer’s instructions Cells

expres-sing recombinant proteins were resuspended in 20 mL

50 mMTris/HCl, pH 7.5, and frozen at)20 C Cells were

thawed in ice-cold water and sonicated Cell lysates were

centrifuged, and supernatants subjected to gel-filtration

chromatography on a Sephadex G75 column (1.8· 90 cm)

Fractions containing purified recombinant proteins were

pooled and stored at)20 C until use

The purity of the recombinant proteins was confirmed by

RP-HPLC using a Wakosil 5C4 column (4.6 mm· 20 cm;

Wako) pre-equlibrated in 0.1% trifluoroacetic acid Bound

proteins were eluted with a linear gradient of 0–100%

acetonitrile/0.1% trifluoroacetic acid The molecular masses

of purified recombinant madanin 1 and 2 were determined

on a Voyager MALDI-TOF mass spectrometer (PerSeptive Biosystems) with a 337-nm N2laser and ion reflector

Assay of effects of madanin 1 and 2 on plasma coagulation

Citrated human normal plasma (20 lL) and recombinant proteins (30 lL) were preincubated for 5 min at 37C Mixtures were activated for 2 min at 37C with 30 lL 25% actin (Dade Behring) in the APTT assay and with 30 lL rabbit brain thromboplastin (Ortho Diagnostic System) in the PT assay Clotting reactions were started by the addition

of 25 lL 50 mMCaCl2, and the clotting time was measured using a coagulometer

Binding analysis using surface plasmon resonance (SPR) SPR measurement was performed using a BIAcore 3000 instrument (BIAcore) Thrombin (bovine) was immobilized

on the surface of a sensor chipCM5 by the amine coupling procedure according to the manufacturer’s instructions Binding analyses were carried out using Hepes-buffered saline (10 mMHep es, p H 7.4, 150 mMNaCl, 5 mMCaCl2, and 0.005% Tween 20) as running buffer at 25C A 40 lL volume of various concentrations of the samples was injected on to the sensor chipat a flow rate of 20 lLÆmin)1 Association was monitored during a 2-min injection of analyte Dissociation was monitored for 2 min after return

to the running buffer Regeneration of the sensor chip surface was achieved with a pulse injection of 1M NaCl The binding data were analyzed using the evaluation software (BIAevaluation) to determine the dissociation constants (Kd)

Assay of effects of madanin 1 and 2 on fibrinogenolytic activity of thrombin

Inhibition of fibrin clot formation by the recombinant proteins was measured using fibrinogen as a substrate Substrate solution was prepared by the addition of 1 part arabic gum (15%, w/v) to 7 parts fibrinogen (100 mg per

7 mL), and preincubated at 37C for 15 min Thrombin (3.9 nM, final concentration) was mixed with various concentrations of recombinant proteins, and the mixtures added to prewarmed substrate solution The prolongation of fibrin clot formation was measured using the coagulometer

Assay of effects of madanin 1 and 2 on activation of factor V and factor VIII by thrombin

The effect of the madanins on the activation of factor V by thrombin was determined as follows Factor V (240 pM, final concentration) in buffer A (50 mMTris/HCl, pH 7.5,

150 mMNaCl, 5 mMCaCl2, and 0.1% BSA) was preincu-bated for 2 min at 37C with thrombin (20 pM, final concentration) in the presence of various concentrations of the recombinant proteins and added to buffer A containing

400 nM prothrombin, 20 pM factor Xa, and 40 lM phos-pholipid After the addition of thrombin and the recom-binant proteins, the reaction mixtures were incubated for

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5 min at room temperature Prothrombin activation by

prothrombinase was stopped by the addition of EDTA

(5 mM, final concentration) The activity of generated

thrombin, which reflects the amount of activated factor V

in the sample, was measured using chromogenic substrate

S-2238

The effect of the madanins on the activation of factor

VIII by thrombin was determined as follows Factor VIII

(0.15 UÆmL)1, final concentration) in buffer A was

pre-incubated for 5 min at 37C with thrombin (2.5 pM, final

concentration) and recombinant proteins, then added to

buffer A containing 400 nMfactor X, 1 nMfactor IXa, and

40 lMphospholipid The reaction mixtures were incubated

for 2 min at room temperature After the incubation, the

activity of factor Xa, which reflects the amount of activated

factor VIII, was measured using chromogenic substrate

S-2222

Assay of effects of madanin 1 and 2 on

thrombin-induced platelet aggregation

Washed platelets were prepared as follows Blood was

mixed with acid citrate dextrose, incubated for 30 min at

room temperature, and centrifuged at 300 g for 10 min

Prostaglandin E1 (PGE1) was added to the supernatant

to a final concentration of 20 ngÆmL)1 The mixture was incubated for 15 min at room temperature and centrifuged

at 1300 g for 20 min The precipitated platelets were washed three times with a modified Tyrode’s buffer (134 mMNaCl,

3 mM KCl, 0.3 mM NaH2PO4, 2 mM MgCl2, 12 mM

NaHCO3, 5 mMglucose, 5 mMHepes, 3.5 mgÆmL)1BSA,

1 mMEGTA) containing 20 ngÆmL)1prostaglandin E1and

20 ngÆmL)1apyrase The resulting platelets were suspended with Tyrode’s buffer containing 2 mMCaCl2

Inhibition of thrombin-induced platelet aggregation was measured using washed platelets Briefly, 550 lL washed platelets (3· 105 platelets per lL) in Tyrode’s buffer containing 2 mM CaCl2, 0.2 mgÆmL)1 fibrinogen, and 1.0 mM Gly-Pro-Arg-Pro peptide was preincubated at

37C for 3 min Then 50 lL of a mixture containing thrombin (0.1 nM, final concentration) and recombinant proteins was added to prewarmed washed platelets Platelet aggregation was monitored using an aggregometer Results

cDNA cloning and expression of madanin 1 and 2 Three distinct cDNA libraries were constructed from the salivary glands of H longicornis at different feeding stages:

1-GCTTTGACCGCAATGAAGCACTTCGCAATTTTGATTCTTGCTGTTGTGGCCAGTGCCGTG

- M K H F A I L I L A V V A S A V

61-GTGATGGCATACCCGGAGAGAGATTCAGCGAAGGAGGGCAACCAAGAGCAAGAGAGAGCT

-V M A Y P E R D S A K E G N Q E Q E R A

121-CTGCATGTAAAGGTACAAAAACGTACTGATGGTGATGCTGACTACGATGAATATGAGGAA

-L H V K V Q K R T D G D A D Y D E Y E E

181-GATGGGACGACTCCTACTCCGGATCCAACTGCACCAACTGCTAAACCACGGCTTCGAGGA

-D G T T P T P D P T A P T A K P R L R G

241-AATAAGCCTTGAATCAATGATGTTCTATTTTTTATAGCGTCCCGATGGCGGTGATGTTGT

-N K P *

301-AGGCTGGAAGCAAATAAAAATACGAAGAGTGACTTCAAAAAAAAAAAAAAAAAAAAAAAA

A

1-GCTTTGACGGCAATGAAGCACTTCGTAATTTTGATTCTTGCTGTTGTGGCCAGTGCCGTG

M K H F V I L I L A V V A S A V

61-GTGATGGCATACCCGGAGAGAGATTCAGCAAAGGACGGCAACCAAGAGAAAGAGAGAGCT

V M A Y P E R D S A K D G N Q E K E R A

121-CTGCTAGTTAAAGTACAAGAACGCTATCAAGGTAATCAAGGTGACTACGATGAATATGAC

L L V K V Q E R Y Q G N Q G D Y D E Y D

181-CAAGATGAGACCACTCCTCCTCCGGATCCAACTGCACAAACTGCAAGACCACGGCTTCGA

Q D E T T P P P D P T A Q T A R P R L R

241-CAAAATCAGGATTGAATCAATGGTGTTCTAGATTTCTATAACCTACCGACGGCGGCAATT

Q N Q D *

301-TTGTGGGGTCCAAACAAATAAAACTACAAAGTGGGACCTCAAAAAAAAAAAAAAAAAAAA

B

Madanin-1 MKHFAILILAVVASAVVMAYPERDSAKEGNQEQERALHVKVQKRTDG-DADYDEYEEDGT

Madanin-2 MKHFVILILAVVASAVVMAYPERDSAKDGNQEKERALLVKVQERYQGNQGDYDEYDQDET

**** ********************** **** **** **** * * ***** * *

Madanin-1 TPTPDPTAPTAKPRLRGNKP

Madanin-2 TPPPDPTAQTARPRLRQNQD

** ***** ** **** *

C

Fig 1 Nucleotide sequences and deduced amino-acid sequences of madanin 1 and 2 The first 19 amino acids are predicted to be the signal peptide sequences for both madanin 1 (A) and madanin 2 (B) (C) Comparison of amino-acid sequences of madanin 1 and 2 Sequence alignment was performed using the ClustalW program at the Bioinformatics Center Institute for Chemical Research The same amino-acid residues are indicated by stars.

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unfed, slow feeding, and rapid feeding A total of 1889

cDNA clones were picked from three cDNA libraries, and

their partial nucleotide sequences determined Sequence

similarity searches of all cDNA clones were performed using

theBLAST program A signal peptide prediction was also

carried out using the SignalP Program, because many

physiologically active molecules identified from the salivary

gland are secreted proteins Predicted secreted proteins were

classified into several protein families based on amino-acid

sequence similarities (data not shown)

The major protein family found in the rapid feeding stage

cDNA library was named the madanin family after the

Jap anese name for H longicornis, Hutatoge-chimadani

The madanin family consists of two proteins, madanin 1

and 2 (Fig 1A,B) They exhibited no sequence similarities

to any other previously identified proteins and shared 79%

sequence identity (Fig 1C) The cDNA of madanin 1 and 2

contained 240 and 243 bpORFs encoding 79 and 80

amino-acids residues, respectively The first 19 amino acids

in both were predicted to be the signal peptide The

calculated molecular mass of the mature regions of madanin

1 and 2 were 6770.9 Da and 7122.42 Da, respectively

To investigate the biological activities of madanin 1 and 2,

the recombinant molecules were produced in Escherichia

coliBL21(DE3) cells using expression vector pET22b Their

expression was confirmed by SDS/PAGE The recombinant

proteins were purified by gel-filtration chromatography

using Sephadex G75, and purity was evaluated by

RP-HPLC (data not shown) The MALDI spectrum of

madanin 1 and 2 exhibited main [M + H]+ions of m/z

6899.39 and 7244.46, respectively (data not shown) As a

methionine residue was added to the N-terminus of the

recombinant proteins when produced using pET22b, the

experimental and calculated masses were almost identical

Madanin 1 and 2 are novel anticoagulants

ofH longicornis

It was previously reported that anticoagulants are present in

the salivary glands of H longicornis [24] According to this

report, an extract of the salivary glands prolonged both

APTT and PT, suggesting the presence of an inhibitor of

either factor Xa or thrombin However, the anticoagulant

molecule has not yet been identified To investigate the

physiological function of madanin 1 and 2, we first

examined their antihemostatic activities using normal

human plasma As shown in Fig 2, madanin 1 and 2

prolonged both APTT and PT in a dose-dependent manner,

demonstrating that they are novel anticoagulants in H

lon-gicornisand inhibit both the intrinsic and extrinsic

coagu-lation pathways

Thrombin is a target molecule of madanin 1 and 2

The results obtained from the APTT and PT assays suggest

that madanins act as inhibitors of factor Xa and/or

thrombin Thus, we investigated, by SPR using BIAcore,

their ability to bind to factor Xa and thrombin All assays

were performed twice with immobilized 821.8 resonance

units (RU) factor Xa or 1037.0 RU thrombin on sensor

chips, as described in Experimental Procedures This assay

clearly showed that madanin 1 and 2 specifically interacted

with thrombin, but not with factor Xa Both bound thrombin in a dose-dependent manner, as shown in Fig 3 From these results, it is clear that thrombin is a target molecule of madanin 1 and 2 However, the sensorgrams showed abnormal patterns, indicating poor interaction, in which the dissociation of the complex was very fast and the reaction between ligand and analyte equilibrated very rapidly

As the association and dissociation phases in the inter-actions of madanin 1 and 2 with thrombin were very short,

it was difficult to analyze the kinetic constants for the inter-action Therefore, we evaluated the equilibrium constants

Fig 2 Effect of madanin 1 and 2 on prolongation of APTT and PT The effects of madanin 1 and 2 on intrinsic and extrinsic pathways were investigated by APTT (A) and PT (B) assays, respectively Various concentrations of madanin 1 and 2 were incubated with citrated human plasma, and the mixture was activated with diluted APTT and

PT reagents After activation, CaCl 2 was added to the mixture, and clotting times were measured (s) Madanin 1; (d) madanin 2.

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usingBIAEVALUATIONsoftware according to the following

equation:

Req¼ KaC=ð1 þ KaCÞ where Reqis the value of resonance units in an equilibrated

state, Kais the association constant, and C is the

concen-tration of the analyte used in the assay Kdwas derived from

the relationship, Kd¼ 1/Ka Under the conditions of this

experiment, Kdvalues for binding of madanin 1 and 2 to the

immobilized thrombin were 4.18 and 2.96 lM, respectively

The two madanins had similar potencies for interaction with

thrombin

Madanin 1 and 2 inhibit the conversion of fibrinogen

into fibrin by thrombin

Next, we examined whether madanin 1 and 2 inhibit the

amidolytic activity of thrombin Assays were performed

using a synthetic substrate for thrombin (S-2238) Thrombin activity remained fully intact despite a 1000-fold molar excess of madanin 1 and 2 added to thrombin, indicating that they do not inhibit amidolytic activity of thrombin with

a small chromogenic substrate (data not shown)

Although madanin 1 and 2 were found by SPR meas-urements to bind to thrombin, they did not inhibit hydrolysis of small synthetic substrates by thrombin Therefore, we examined whether they inhibit cleavage of the physiological substrate, fibrinogen As shown in Fig 4, they prolonged fibrin clot formation by thrombin in a dose-dependent manner, showing that they prevented thrombin from cleaving fibrinogen and are able to inhibit the function

of thrombin without affecting its amidolytic activity The results of these two experiments suggest that thrombin inhibition by madanin 1 and 2 is caused by competitive binding to the fibrinogen-binding site (anion-binding exo-site 1) on the thrombin molecule, and not from binding to the active site

Fig 3 SPR analysis of the interaction between madanin 1 and 2 and thrombin Interactions between thrombin and madanin 1 (A) and madanin 2 (B) were investigated by SPR measurement Thrombin was immobilized on

a sensor chipat levels of 1037.0 RU Sensor-grams were obtained by injection of madanin

1 and 2 at different concentrations ranging from 0.25 l M to 5 l M at a flow rate of

20 lLÆmin)1and are indicated as solid lines The sensor chipsurface was regenerated with

1 M NaCl before each injection The inter-actions between factor Xa and madanin 1 and 2 were also investigated Factor Xa was coupled

to a sensor chipat levels of 821.8 RU The interactions were measured by injection of

5 l M madanin 1 and 2 The sensorgrams are shown as dotted lines in each case.

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Madanin 1 and 2 inhibit activation of factors V

and VIII and aggregation of platelets by thrombin

It has been reported that the anion-binding exosite 1 is

involved in molecular recognition of thrombin for factor V,

factor VIII, and PAR on platelets Therefore, we

investi-gated the effects of madanin 1 and 2 on thrombin-catalyzed

activation of factors V and VIII and thrombin-induced

aggregation of platelets As shown in Figs 5 and 6,

activation of prothrombin and factor X by prothrombinase

and tenase were inhibited, respectively, by the presence of

madanin 1 and 2 It was further confirmed that they do not

inhibit the activity of prothrombinase itself The activities of

thrombin and factor Xa in each assay reflect the amount of

factors Va and VIIIa, respectively, present Thus, it is

indicated that madanin 1 and 2 inhibit activation of factors

V and VIII by thrombin and then prevent these cofactors

from forming prothrombinase and tenase complexes On

the other hand, as shown in Fig 7, madanin 1 and 2

suppressed thrombin-induced aggregation of platelets in a

dose-dependent manner Platelet aggregation begins with

proteolytic cleavage of PAR by thrombin Therefore, these

results also support the conclusion that madanin 1 and 2 are

direct competitive inhibitors of the anion-binding exosite 1

on thrombin

Discussion

In this study, we have identified novel anticoagulants,

named madanin 1 and 2, from the salivary gland of

H longicornis This is the first description of antihemostatic

factors in H longicornis Madanin 1 and 2 prolonged both

APTT and PT, indicating that they are anticoagulants for

the common pathway of coagulation SPR analysis showed that they specifically interacted with thrombin, not with factor Xa An inhibition assay with fibrinogen as substrate showed that they inhibited the fibrinogenolytic activity of thrombin Therefore, we conclude that they inhibit blood coagulation by inhibiting the function of thrombin Madanin 1 and 2 also inhibited the activation of thrombin-catalyzed cofactors V and VIII as well as the conversion of fibrinogen into fibrin Furthermore, they inhibited thrombin-induced platelet aggregation via proteo-lytic activation of PAR However, they did not inhibit the amidolytic activity of thrombin in an assay using a synthetic

Fig 4 Effect of madanin 1 and 2 on formation of fibrin clot by thrombin.

Substrate solution containing fibrinogen and arabic gum was prepared

as described in Experimental Procedures and prewarmed at 37 C.

Thrombin and various concentrations of madanin 1 and 2 were mixed

and added to the substrate solution The time to fibrin clot formation

was measured using a coagulometer (s) Madanin 1; (d) madanin 2.

Fig 5 Effect of madanin 1 and 2 on the activation of factor V by thrombin Factor V was preincubated with thrombin in the presence of madanin 1 or 2 and added to buffer A containing prothrombin (400 n M ), factor Xa (20 p M ), phospholipid (40 l M ), and CaCl 2 (5 m M ) Thrombin activity was measured using a chromogenic sub-strate (S-2238).

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substrate It has been reported that fibrinogen and PAR

bind to the anion-binding exosite 1 on thrombin and that

anion-binding exosite 1 and 2 are involved in the

inter-actions between thrombin and its cofactors Structural data

on thrombin show that anion-binding exosite 1 and 2 are

located opposite each other on the thrombin molecule [25–

27] Taking the inhibitory profiles and molecular sizes of

madanin 1 and 2 into consideration, it is most likely that

they are competitive inhibitors directed to the anion-binding

exosite 1 of thrombin

Madanin 1 and 2 show no similarities in their amino-acid sequences to thrombin inhibitors from any blood-sucking organisms However, the clusters of acidic residues found in the central regions of madanin are similar to those found in hirudin [17], tsetse thrombin inhibitor [28,29], anophelin [30,31], and thrombostatin [32] These acidic regions show electrostatic interactions with positively charged anion-binding exosite 1 Furthermore, our recent studies indicate that N-terminally truncated madanin 1 maintains the ability

to bind to thrombin (unpublished data) Thus, madanin 1 and 2 may bind to anion-binding exosite 1 through the acidic residue clusters

The Kdvalues of madanin 1 and 2 determined by SPR analysis (4.18 and 2.96 lM, respectively) are significantly higher than those of other anion-binding exosite 1 inhibitors [17,28,31] In the SPR analysis, thrombin was immobilized

Fig 7 Effect of madanin 1 and 2 on platelet aggregation by thrombin Washed platelets were prepared as described in Experimental Proce-dures Thrombin and the madanins were mixed and added to the washed platelets (3 · 10 5 platelets per lL) in the presence of 2 m M

CaCl 2 , 0.2 mgÆmL)1fibrinogen, and 1.0 m M Gly-Pro-Arg-Pro peptide Platelet aggregation was monitored with an aggregometer Complete aggregation was obtained in the absence of madanin 1 and 2 Fig 6 Effect of madanin 1 and 2 on activation of factor VIII by

thrombin Factor VIII was preincubated with thrombin in the presence

of madanin 1 or 2 and added to buffer A containing factor X (400 n M ),

factor IXa (1 n M ), phospholipid (40 l M ), and CaCl 2 (5 m M ) Factor

Xa activity was measured using a chromogenic substrate (S-2222).

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by amine coupling In this immobilizing reaction, the

e-amino groupof the lysine residue on the anion-binding

exosite 1 of thrombin may be coupled to the carboxy group

on the sensor chip This may result in the weak interactions

observed In fact, the Kd(app) values calculated from the

results in Figs 5 and 6 by the methods of Henderson [33] are

85–170-fold lower than those obtained from SPR analysis

The Kd(app)values of madanin 1 and 2 are 25 and 34.5 nM,

respectively Therefore, it is possible that the Kdvalues of

interactions between madanins and thrombin are lower

than those determined by SPR analysis

Physiological coagulation is initiated by formation of a

factor VIIa–tissue factor complex at the injury site This

complex activates factor X, which is followed by generation

of small amounts of thrombin [34] The generated thrombin

further activates factor V and factor VIII, which leads to the

generation of a large amount of thrombin This

amplifica-tion stepis thought to be crucial for physiological

coagu-lation, as deficiencies of factor V and factor VIII cause

serious bleeding Therefore, it is possible that madanins

inhibit blood coagulation at this initial stepby inhibiting the

activation of factor V and factor VIII by thrombin and

contribute considerably to tick blood feeding

In conclusion, novel thrombin inhibitors (madanin 1 and 2)

from H longicornis have been identified They inhibit

various physiological functions of thrombin without

inter-fering with its catalytic activity and may play an important

role in tick blood feeding Recent studies report that

anion-binding exosite 1 inhibitors such as the C-terminal peptide of

hirudin are attractive therapeutic drugs for arterial

throm-bosis [35] Thus, further studies on the inhibitory mechanisms

of madanin 1 and 2 may provide useful information for the

development of therapeutic agents for thrombosis

Acknowledgements

This work was supported by a grant from the Japan Society for

Promoting Science: Future Developmental Research (to Y C.) and by

a grant from Mitsubishi Pharma Research Foundation (to S I.).

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