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MSP is synthesized and secreted by hepatocytes [6] and circu-lates in plasma as a single-chain precursor pro-MSP Keywords activation; hepatocyte growth factor activator; macrophage-stimu

Trang 1

of single-chain precursor macrophage-stimulating protein Makiko Kawaguchi, Hiroshi Orikawa, Takashi Baba, Tsuyoshi Fukushima and Hiroaki Kataoka Section of Oncopathology and Regenerative Biology, Department of Pathology, Faculty of Medicine, University of Miyazaki, Japan

Macrophage-stimulating protein (MSP) was originally

identified as a plasma protein that promotes

chemotac-tic responses in peritoneal resident macrophages [1–3]

Mature MSP is a disulfide-linked heterodimer with a

relative molecular mass of 80–95 kDa, consisting of an

a chain of approximately 60 kDa and a b chain of

approximately 30 kDa, that autophosphorylates its

specific receptor tyrosine kinase RON (recepteur

d’origine nantais) [4,5] MSP is a member of the krin-gle proteins, which contain multiple copies of a highly conserved triple disulfide loop structure (kringle domain) The a chain contains an N-terminal hairpin loop, followed by four kringle domains, and the b chain has a serine protease-like domain [5] MSP is synthesized and secreted by hepatocytes [6] and circu-lates in plasma as a single-chain precursor (pro-MSP)

Keywords

activation; hepatocyte growth factor

activator; macrophage-stimulating protein;

recepteur d’origine nantais (RON); serum

Correspondence

H Kataoka, Section of Oncopathology and

Regenerative Biology, Department of

Pathology, Faculty of Medicine, University

of Miyazaki, 5200 Kihara, Kiyotake, Miyazaki

889-1692, Japan

Fax: +81 985 85 6003

Tel: +81 985 85 2809

E-mail: mejina@med.miyazaki-u.ac.jp

(Received 14 February 2009, revised 7 April

2009, accepted 22 April 2009)

doi:10.1111/j.1742-4658.2009.07070.x

Macrophage-stimulating protein (MSP) is a plasma protein that circu-lates as a single-chain proform It acquires biological activity after prote-olytic cleavage at the Arg483–Val484 bond, a process in which serum and cell surface serine proteinases have been implicated In this article,

we report that hepatocyte growth factor activator (HGFA), a serum proteinase which activates hepatocyte growth factor in response to tissue injury, may have a critical role in the activation of pro-MSP In vitro analysis has revealed that human HGFA efficiently cleaves human pro-MSP at the physiological activation site without further degradation, resulting in biologically active MSP, as measured by the chemotactic response and MSP-induced morphological change of peritoneal macro-phages The processing of pro-MSP by HGFA is 10-fold more efficient than processing by factor XIa To search for a role of HGFA in pro-MSP activation, we analyzed the processing of mouse pro-pro-MSP in sera from HGFA-knockout (HGFA)/)) mice The proform of MSP was the predominant molecular form in the plasma of both wild-type and HGFA)/) mice In wild-type sera, endogenous pro-MSP was progres-sively converted to the mature two-chain form during incubation at

37C However, this conversion was significantly impaired in sera from HGFA)/) mice The addition of recombinant HGFA to HGFA-deficient serum restored pro-MSP convertase activity in a dose-dependent manner, and a neutralizing antibody to HGFA significantly reduced the conver-sion of pro-MSP in wild-type serum Moreover, initial infiltration of macrophages into the site of mechanical skin injury was delayed in HGFA)/) mice We suggest that HGFA is a major serum activator of pro-MSP

Abbreviations

CHO, Chinese hamster ovary; HGF/SF, hepatocyte growth factor/scatter factor; HGFA, hepatocyte growth factor activator; LPS,

lipopolysaccharide; MSP, macrophage-stimulating protein; PC50%, processing concentration 50%; PCI, protein C inhibitor; RON, recepteur d’origine nantais.

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that has no biological activity until the protein is

cleaved into a and b chains at the Arg483–Val484

bond [5] Several proteinases have been identified as

candidate convertases in the processing of pro-MSP to

mature MSP Of interest is the observation that

pro-MSP activation occurs in the presence of fetal bovine

serum in vitro, suggesting the existence of a pro-MSP

convertase in serum [7] However, this conversion was

not observed in freshly prepared human serum [8] As

pro-MSP is abundant in plasma (2–5 nm) [5],

activation of pro-MSP by the serum convertase may

be an important physiological response to tissue injury

Previous studies have suggested that the proteinases

involved in the coagulation cascade and inflammation,

such as factor XIa, factor XIIa and serum kallikrein,

are responsible for pro-MSP convertase activity

in serum [7] However, the physiological serum

activator of pro-MSP remains to be determined

Membrane-bound serine proteinases are also important

[8] Matriptase/ST14 may be an important cellular

activator of pro-MSP in the pericellular

microenviron-ment [9] Other potential activators of pro-MSP

include mouse epidermal growth factor-binding protein

and nerve growth factor c (kallikrein 1-related

peptidase b3), both of which have serine proteinase

activity [10]

Hepatocyte growth factor/scatter factor (HGF/SF)

is also a member of the kringle protein family and

shows significant sequence homology to MSP (45%

amino acid sequence identity) [2,3,11] Like MSP,

HGF/SF is secreted as an inactive single-chain

precur-sor (pro-HGF/SF), and the cleavage between Arg494

and Val495 by an extracellular proteinase is critical for

signal transduction via its specific cell surface receptor

tyrosine kinase, MET, the protein product of the c-met

proto-oncogene [12] The serine proteinase hepatocyte

growth factor activator (HGFA) is a very efficient

pro-cessor of pro-HGF/SF [12,13] HGFA is synthesized

by the liver and circulates as an inactive zymogen

(pro-HGFA) at a concentration of approximately

40 nm [14] It is activated in response to tissue injury

via cleavage of the bond between Arg407 and Ile408,

resulting in a two-chain heterodimeric active form

[12,15] This cleavage is assumed to be mediated by

thrombin in the serum and by kallikrein 1-related

peptidases, such as KLK4 and KLK5, in the

pericellu-lar microenvironment [16,17] Activated serum HGFA

retains sufficient activity in bovine serum and also

in mouse serum [12,18] However, in human

serum, its activity is inhibited by protein C inhibitor

(PCI) [14] The activity of HGFA is also regulated by

a cell surface inhibitor, namely HGFA inhibitor, in

local tissues [19]

Considering the significant structural similarity of MSP to HGF/SF, we hypothesized that HGFA, a serum activator of pro-HGF/SF, may be an important candidate for the serum pro-MSP convertase in vivo

188

188

62

49

38

38

28

49

62

28

L ys Leu Arg V a l V a l Gly Gly His Pro

483 484

anti-MSP anti-His tag

0 0.005 0.05 0.5 5 0 0.005 0.05 0.5 5 10 20

pro-MSP

α chain

HGFA (n M ) Factor XIa (n M )

0 5 10 30 60 120 0 5 10 30 60 120

pro-MSP

α chain

HGFA (0.5 n M ) Factor XIa (0.5 n M )

Incubation time (min)

0 0.05 0.5 5 0 0.05 0.5 5 0 0.05 0.5 5 0 10 20 10 20

HGFA (n M ) Factor XIa (n M )

pro-MSP

α chain

NaCl concentration (m M )

A

B

C

D

Fig 1 Processing of pro-MSP by HGFA (A) Immunoblot analysis

of proteolytic cleavage of a His-tagged human pro-MSP recombi-nant protein by human HGFA Pro-MSP at a concentration of 5 n M was incubated with 0.5 n M of HGFA in 20 m M Tris buffer (pH 7.6),

150 m M NaCl and 0.05% Chaps for 8 h at 37 C Anti-MSP IgG rec-ognized the a chain of MSP and the anti-His tag IgG recrec-ognized the poly-His tag at the C-terminus of MSP The N-terminal amino acid sequence of a product of approximately 30 kDa is indicated (B) Effects of NaCl concentration on the processing of pro-MSP Pro-MSP at a concentration of 5 n M was incubated with various concentrations of HGFA or factor XIa in 20 m M Tris buffer (pH 7.6) and 0.05% Chaps, with 50–150 m M of NaCl, for 4 h at 37 C The processed products were analyzed by immunoblot (C) Dose-depen-dent processing of pro-MSP (5 n M ) by HGFA or factor XIa in Tris buffer (pH 7.6), 50 m M NaCl and 0.05% Chaps The reaction mix-tures were incubated for 4 h at 37 C (D) Time-dependent process-ing of pro-MSP (5 n M ) by HGFA (0.5 n M ) or factor XIa (0.5 n M ) in Tris buffer (pH 7.6), 50 m M NaCl and 0.05% Chaps.

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In this study, we found that recombinant human

HGFA efficiently converts human pro-MSP to its

active form in vitro Subsequent experiments using an

HGFA-deficient mouse model [18] revealed that

HGFA is a major serum activator of pro-MSP

Results

Processing of pro-MSP by HGFA

The effect of recombinant human HGFA was tested

on the processing of recombinant human pro-MSP

Incubation of pro-MSP with different concentrations

of HGFA at 37C led to the processing of pro-MSP

in a dose-dependent manner Immunoblot analysis

using an anti-MSP IgG revealed a band of

approxi-mately 60 kDa, presumably the a chain of mature

MSP (Fig 1A) Generation of a band of

approxi-mately 30 kDa, presumably the b chain, was also

detected by an anti-His tag IgG (Fig 1A) Cleavage

site analysis was performed after separating the

products of HGFA cleavage by SDS–PAGE under

reducing conditions The N-terminal amino acid

sequence of the 30 kDa product was

Val-Val-Gly-Gly-His Therefore, this 30 kDa band was in fact the b

chain of mature MSP, and HGFA cleaved pro-MSP at

the normal processing site, Arg483–Val484 (Fig 1A)

The processing was suppressed at higher

concentra-tions of NaCl, and this tendency was also observed for

factor XIa, a known serum activator of pro-MSP

(Fig 1B) The concentration of HGFA required to acti-vate 50% of 5 nm pro-MSP (PC50%) after 4 h at 37C was 0.05 nm, whereas that of factor XIa was 0.5 nm Therefore, HGFA was a 10-fold more potent convertase

of pro-MSP than factor XIa (Fig 1C) Further degra-dation of mature MSP was not observed by HGFA We also examined the time course of pro-MSP processing

by HGFA (Fig 1D) More than 50% of pro-MSP (5 nm) was processed within 30 min by 0.5 nm of HGFA, again showing superior efficiency to factor XIa

Biological activity of MSP processed by HGFA The biological activity of MSP after HGFA processing was determined using macrophage chemotaxis assays Pro-MSP could not efficiently induce the chemotactic migration of macrophages However, after incubation

of pro-MSP with HGFA, the processed products showed a significant induction of macrophage migra-tion, and the activity was comparable with that of commercially available recombinant mature human MSP a/b heterodimer (Fig 2A) Macrophages derived from HGFA)/) mice also responded to the recombi-nant mature MSP a/b heterodimer (data not shown) HGFA alone did not detectably induce the chemotac-tic response We also examined the effect of HGFA processing on the culture morphology of mouse perito-neal macrophages The MSP processed by HGFA induced an elongated, migratory morphology of macrophages within 1 h, showing an effect similar to

0

20

80

+

pro-MSP

HGFA

MSP

40

60

*

*

No treatment + MSP

+ pro-MSP + HGFA-treated pro-MSP Control

pro-MSP

pro-MSP + HGFA Fig 2 Biological activity of MSP processed by HGFA (A) Results of chemotaxis assays Murine peritoneal resident macrophages (1 · 10 5 cells) were placed in the upper well of Chemotaxicells and incubated for 3.5 h at 37 C The bottom well contained pro-MSP (1.25 n M ) with or without HGFA (0.125 n M ) pretreatment (2 h) or recombinant active MSP (1.25 n M ) Values are the mean number ± stan-dard deviation of migrated cells per high-power field in triplicate experiments *P < 0.01 compared with control (pro-MSP only, HGFA only or

no addition) (Mann–Whitney U-test) Representative photographs of migrating cells are also shown (B) Morphology of macrophages in the presence of pro-MSP (1.25 n M ), MSP (1.25 n M ) or pro-MSP (1.25 n M ) pretreated with HGFA (0.125 n M ) After 1 h in culture, the cells were observed by phase-contrast microscopy.

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that of the recombinant mature MSP a/b heterodimer

(Fig 2B)

Impaired processing of endogenous pro-MSP

in serum from HGFA-/-mice

In order to study further the role of HGFA in the

acti-vation of pro-MSP, we used HGFA)/) mice After

incubation at 37C for 2 h, most of the endogenous

MSP proteins in the plasma from both wild-type and

HGFA)/) mice were the 90 kDa single-chain

pro-forms (Fig 3A) In contrast, pro-MSP was apparently

processed in the sera from wild-type mice after a 2 h

incubation at 37C (Fig 3B), indicating the presence

of pro-MSP convertase in the wild-type serum, as

pre-viously observed in bovine serum [7] However, the

processing of pro-MSP was reduced significantly in the sera from HGFA)/)mice (Fig 3B) A subsequent time course study also confirmed the significantly reduced processing activity of pro-MSP in HGFA-deficient sera relative to that in wild-type sera (n = 5 for each group) (Fig 3C) Although the processing of endo-genous pro-MSP was apparent within 15 min of incu-bation and had reached 20% at 30 min in wild-type serum, there was less than 10% processing even after

120 min of incubation in HGFA-deficient serum (Fig 3C) Therefore, the absence of HGFA resulted in

a markedly delayed and reduced processing of pro-MSP in mouse serum Indeed, the addition of recombi-nant HGFA to the sera of HGFA)/) mice restored the pro-MSP processing activity (Fig 4)

Inhibition of pro-MSP processing activity

in serum by anti-HGFA neutralizing IgG The efficient pro-MSP activating activity of human HGFA in vitro and the significantly reduced processing

of endogenous pro-MSP in HGFA-deficient mouse serum suggest that HGFA is a major serum activator

of pro-MSP in vivo Therefore, we examined the effect

of a neutralizing antibody raised against HGFA (P1-4)

on the pro-MSP convertase activity of wild-type mouse serum The P1-4 antibody suppressed significantly the processing of pro-MSP in sera obtained from wild-type mice (Fig 5) We concluded that HGFA is a major activator of pro-MSP in mouse serum

Delayed infiltration of macrophages in HGFA -/-mice at a site of tissue injury

To test the physiological role of HGFA-mediated acti-vation of pro-MSP, we compared the recruitment of macrophages in injured tissues, in which the activation

of pro-HGFA was anticipated by thrombin and/or

0

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30

40

50

60

70

pro-MSP

pro-MSP

pro-MSP

α-chain

α-chain

α-chain

120 min

120 min

60 min

Incubation

time

Incubation

time

Plasma

A

B

C

Serum

HGFA–/–

Wild

Incubation time (min)

Wild-type serum HGFA-deficient serum

*

Fig 3 Impaired processing of endogenous pro-MSP in

HGFA-defi-cient serum (A) Processing of endogenous pro-MSP in plasma

from wild-type and HGFA)/)mice Plasma was incubated at 37 C

and the processing of endogenous pro-MSP was analyzed by

immunoblot using anti-MSP antibody (B) Processing of

endo-genous pro-MSP in sera from wild-type and HGFA)/)mice Serum

was incubated at 37 C and the processing of endogenous

pro-MSP was analyzed by immunoblot (C) Time course of pro-pro-MSP

processing in serum Values are the mean processing rate ±

stan-dard deviation (n = 5) *P < 0.001, Mann–Whitney U-test.

Incubation: 120 min

HGFA (nM)

% converted pro-MSP

pro-MSP MSP α chain

Fig 4 Reversion of pro-MSP convertase activity in serum from HGFA)/) mice by recombinant HGFA HGFA-deficient serum was incubated with recombinant human HGFA (0–180 n M ) for 2 h, and the processing of endogenous pro-MSP was analyzed by immunoblot.

Trang 5

KLKs, between wild-type mice and HGFA)/) mice.

We generated a small mechanical wound in the dorsal

skin of mice and examined the infiltration of

macro-phages by measuring the CD68 mRNA level One day

after injury, the levels of CD68 mRNA in the wounds

of HGFA)/) mice were significantly lower than those

of wild-type mice (Fig 6A) The level of MSP

pro-cessing was also low in HGFA)/) wounds (Fig 6B)

However, at the fifth day of injury, the CD68 mRNA

level was comparable between HGFA)/) wounds and

wild-type wounds (Fig 6A) These results suggest that

serum HGFA is required for the early-phase

recruit-ment of macrophages at the injured tissue, possibly via

its efficient pro-MSP processing activity

Discussion

Pro-MSP is primarily produced by the liver [6] and

cir-culates in blood with a concentration of 2–5 nm [5] It is

converted to its mature active form during blood

coagu-lation and local inflammation [5,7] Wound fluids also

contain pro-MSP convertase activity, and a cellular

sur-face proteinase is also an important convertase [5,8,9]

This activation step of pro-MSP might serve as a critical

regulatory mechanism in MSP-induced physiological

and pathophysiological tissue responses After

proteo-lytic cleavage, it stimulates resident macrophages via its

specific receptor tyrosine kinase, RON [5,11] Epithelial

cells and neoplastic cells also frequently express RON

[11,20,21] The establishment of RON-induced signaling

appears to have an important role in inflammatory

pro-cesses [1,5,22–24], cellular survival and wound healing

[25,26] It is also important in the progression and

meta-static spread of various types of tumor [11,27,28] In this

study, we have shown that human HGFA efficiently

activates human pro-MSP in vitro In mice, serum

HGFA represents the major pro-MSP convertase

activ-ity of the serum Indeed, the conversion of endogenous

pro-MSP to its mature form was impaired in sera from

HGFA)/)mice and the convertase activity in wild-type sera was significantly attenuated by the addition of anti-HGFA neutralizing IgG Moreover, initial infiltration

of macrophages into the site of mechanical skin injury was delayed in HGFA)/) mice Together with the fact that matriptase, a cell surface activator of pro-MSP [9],

is also a potent activator of pro-HGF/SF [29], we sug-gest that pro-MSP might share its activation machiner-ies with pro-HGF/SF (Fig 7)

The identification of HGFA as a major serum activa-tor of pro-MSP may explain why the serum convertase activity of pro-MSP is different between species Although bovine serum [7] and mouse serum showed significant processing activity for endogenous pro-MSP, the processing activity of human serum was very weak and the molecular form of MSP in human serum was mostly proforms (data not shown), as described previ-ously [8] HGFA is resistant to major serum proteinase inhibitors [12] and is active in mouse serum [18] How-ever, it can be inhibited by PCI, a serpin-type protein-ase inhibitor present in human plasma [14,30] However, mouse plasma does not contain PCI [30] Therefore, HGFA-mediated conversion of pro-MSP may be tightly regulated by PCI in human serum, whereas HGFA remains active and easily converts pro-MSP in mouse serum because of the absence of PCI HGFA is present in plasma as an inactive zymogen

at a concentration of approximately 40 nm in humans [14] During tissue injury, pro-HGFA is converted to the active heterodimeric form by thrombin [12,15] Human kallikrein 1-related peptidases, KLK4 and KLK5, are also candidate activators of pro-HGFA in the local tissue environment [17] After conversion, mature HGFA very efficiently activates pro-HGF/SF

at the site of injury [16], which might have important roles in survival, repair and regeneration of the injured tissue [12,19] The activity of HGFA is tightly regu-lated by PCI in human serum and also by HGFA inhibitor type 1 and type 2 on the epithelial cell surface [12,14,19] Nonetheless, the activity of HGFA is detectable in injured human tissues, such as invasive tumors, accompanying the activation of pro-HGF/SF [31] To date, the possible involvement of HGFA in tissue repair and cancer progression has been discussed primarily in the context of its presumed capability to activate pro-HGF/SF and the subsequent MET signal-ing cascade [11,12,18] This study indicates that MSP-induced RON signaling can be initiated by HGFA activity and may contribute to the role of HGFA in tissue repair and cancer progression Furthermore, the activation of pro-MSP by HGFA prompts the consid-eration of the possible role of HGFA in inflammation via modulation of macrophage function

IgG1 P1-4 IgG1 P1-4 IgG1 P1-4

Incubation time

Antibody

pro-MSP

MSP α chain

— — — 35 38 14 52 61 29%

% converted

pro-MSP

Fig 5 Inhibition of pro-MSP processing activity in serum by

anti-HGFA IgG Serum from wild-type mice was incubated for the

indicated time periods at 37 C without or with 400 lgÆmL)1 of

anti-HGFA neutralizing IgG (P1-4) or nonspecific mouse IgG1 The

processing of endogenous pro-MSP was analyzed by immunoblot.

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Evidence suggests that MSP exerts a dual function,

both stimulatory and inhibitory, on macrophages [5]

Stimulatory functions include its ability to induce

mac-rophage spreading, migration, phagocytosis and the

production of cytokines [1,5,32] However, MSP

inhib-its lipopolysaccharide (LPS)-induced production of

inflammatory mediators and, consequently, RON-defi-cient mice show increased inflammatory responses and susceptibility to LPS-induced septic death [5,22–24]

Therefore, MSP is also required to attenuate an exces-sive inflammatory response to LPS stimulation, and thus may have an important regulatory role in septic

1.11 ± 0.22 1.08 ± 0.16

CD68

β-actin

Wild-type

A

B

HGFA KO

0 day

0.33 ± 0.08 CD68/actin 0.30 ± 0.06

1.25 ± 0.05 0.79 ± 0.15

*

Wild KO

Wild KO Incubated Serum

pro-MSP MSP α chain

% converted pro-MSP 44 33 — 48 84 < 5 18 — — < 5 —

Fig 6 Delayed infiltration of macrophages in cutaneous wounds of HGFA)/)mice (A) Infiltration of macrophages in wounded skin tissue

was evaluated by CD68 mRNA level *P < 0.05, Mann–Whitney U-test (n = 4) (B) Processing level of pro-MSP in injured tissues (1 day after

injury) Skin tissues without injury (0 day) were also examined For positive control, wild-type serum after incubation for the processing of

endogenous pro-MSP (incubated serum) was also applied Wild, wild-type mice; KO, HGFA)/)mice.

Tissue injury

Activation of coagulation cascade

pro-thrombin Thrombin

pro-HGFA

pro-MSP

MSP

pro-HGF/SF

HGF/SF

RON

MET

HGFA

EGF-BP, NGF-γ

Matriptase

KLK4, KLK5

Cell surface proteinase(s)

Macrophages Epithelial

cells Tumour cells

Endothelial cells

Factor XIa Factor XIIa

Fig 7 Hypothetical model for the activation of pro-MSP There may be diverse pathways for the activation of pro-MSP, and pro-MSP might

share the activation machinery with its homologous protein, pro-HGF/SF One pathway is mediated by membrane-bound serine proteinases

(cell surface activator), such as matriptase [9] Matriptase is also a potent activator of pro-HGF/SF [12,30] The second pathway is mediated

by humoral activators that are generated in injured tissues The activation of the coagulation cascade by tissue injury eventually results in

the active form of HGFA that efficiently activates both pro-MSP and pro-HGF/SF Other coagulation proteinases, such as factor XIa and

fac-tor XIIa, may also mediate the activation of pro-MSP [7] and pro-HGF/SF [12] Wound fluids in the injured tissues contain other pro-MSP

acti-vators, such as epidermal growth factor-binding protein (EGF-BP) and nerve growth factor c (NGF-c) [10] The effects of EGF-BP and NGF-c

on pro-HGF/SF are unknown.

Trang 7

inflammation In the septic condition, intravascular

hypercoagulation occurs, which might result in the

conversion of pro-HGFA to its active form The

HGFA-mediated activation of pro-MSP may be an

important process in the regulation of macrophage

functions in septic inflammatory responses To test this

hypothesis, future studies of HGFA)/)mice under

vari-ous inflammatory stimuli, including LPS stimulation,

will be required Moreover, the difference in serum

HGFA activity between human and mouse may

have implications in the different susceptibility to

LPS-induced septic death between these species

In summary, we have demonstrated for the first time

that HGFA is a potent activator of pro-MSP Although

the activation of pro-MSP is a redundant system which

can be mediated by various proteinases (Fig 7) [7–10],

the major pro-MSP convertase in serum is HGFA As

pro-HGFA is activated in response to tissue injury, we

suggest that HGFA-mediated activation may play an

important role in the regulation of MSP/RON signaling

involved in inflammation, wound healing and cancer

progression Further experiments of tissue injury and

inflammation using genetically engineered mouse

mod-els of the HGFA and MSP genes are needed to explore

the in vivo significance of HGFA in MSP/RON

signal-ing However, our study also indicates that caution

should be exercised when interpreting the function of

MSP/RON signaling using a mouse model in vivo, as

HGFA activity would be much higher in mouse serum

than in human serum because of the absence of

circulat-ing PCI in mice [30]

Experimental procedures

Antibodies

Anti-human MSP goat polyclonal IgG and the recombinant

active form of human MSP were purchased from R&D

Sys-tems (Minneapolis, MN, USA) Recombinant human factor

XIa was obtained from Haematologic Technologies, Inc

polyclonal IgG (T-19) was obtained from Santa Cruz

Biotechnology (Santa Cruz, CA, USA) The preparation of

the recombinant active form of human HGFA and

anti-human HGFA mouse monoclonal neutralizing IgG P1-4,

which is also cross-reactive to mouse HGFA, has been

described previously [15] Anti-His tag rabbit polyclonal IgG

was purchased from MBL (Nagoya, Japan)

Preparation of recombinant proteins

The preparation of the recombinant active form of HGFA

has been described previously [19] To obtain a recombinant

pro-MSP protein, the entire coding region of the MSP gene was subcloned into the pcDNA3.1/myc-HisA expression plasmid (Invitrogen, Carlsbad, CA, USA) and transfected into Chinese hamster ovary (CHO) cells using Lipofectamine

2000 reagent (Invitrogen) After transfection, the cells were cultured in DMEM containing 10% fetal bovine serum and gradually changed to serum-free medium (CHO-S-SFMII; Invitrogen) containing 250 lgÆmL)1 G418 (Sigma-Aldrich,

St Louis, MO, USA) To prevent the cleavage of pro-MSP

by cellular and fetal bovine serum-derived proteases, cells were cultured in the presence of 50 lm nafamostat mesilate (Torii Pharmaceutical Co., Tokyo, Japan) G418-resistant colonies were selected and screened for the expression and production of pro-MSP Supernatants were collected from the serum-free cultures every day and 0.1% Chaps (Sigma-Aldrich) was added Recombinant pro-MSP in the condi-tioned medium was affinity purified with TALON His-Tag Purification Resins (Clontech Laboratories, Mountain View,

CA, USA) according to the manufacturer’s instructions

Activation of pro-MSP

Recombinant pro-MSP (final concentration, 5 nm) was incubated with various concentrations of HGFA or factor XIa in 20 lL reactions containing 20 mm Tris/HCl, 50–150 mm NaCl and 0.05% Chaps (pH 7.6) for the indi-cated time periods at 37C The processing of pro-MSP was determined by immunoblot analysis under reducing conditions, and the extent of processing was verified using photoshopsoftware (Adobe Systems, San Jose, CA, USA) The specific activity for pro-MSP processing was expressed

as the enzyme concentration required for the conversion of 50% of 5 nm pro-MSP to its mature form, and was desig-nated as the processing concentration 50% (PC50%) To assess the time course of cleavage by HGFA or factor XIa, pro-MSP (5 nm) was incubated with 0.5 nm of each proteinase at 37C for various time periods (0–120 min)

Immunoblot analysis

Each sample was mixed with SDS–PAGE sample buffer and heated for 15 min at 70C SDS–PAGE was per-formed under reducing conditions using 4–12% gradient gels After electrophoresis, samples were transferred to Immobilon poly(vinylidene difluoride) membranes (Milli-pore, Bedford, MA, USA) After blocking with 3% BSA in Tris-buffered saline (TBS) with 0.05% Tween-20 (TBS-T), the membranes were incubated with primary antibody at

4C overnight, followed by washing in TBS-T and incuba-tion with a horseradish peroxidase-conjugated rabbit anti-goat IgG (DAKO, Glostrup, Denmark) diluted in TBS-T with 1% BSA for 1 h at room temperature The labeled proteins were visualized with a chemiluminescence reagent (PerkinElmer Life Science, Boston, MA, USA)

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N-terminal amino acid sequencing of cleaved

pro-MSP

Pro-MSP (final concentration, 416 nm) was incubated with

97 nm HGFA in a 40 lL reaction containing 20 mm Tris/

HCl, 150 mm NaCl and 0.05% Chaps (pH 7.6) at 37C for

11 h The reaction mixture was subjected to SDS–PAGE,

after which the proteins were transferred to an Immobilon

membrane and stained with 0.1% Coomassie Brilliant Blue

in a water–methanol–acetic acid solution (4.5 : 4.5 : 1, v/v)

The cleaved MSP protein band was cut and processed for

N-terminal amino acid sequencing by automated Edman

degradation using the Procise 494 HT Protein Sequencing

System (Applied Biosystems, Foster City, CA, USA)

Preparation of peritoneal macrophages and

bioassays

Murine peritoneal resident macrophages were obtained

from C57BL/6 mice by washing the peritoneal cavity with

3 mL per mouse of serum-free RPMI-1640 medium Cells

were washed and resuspended in RPMI-1640 medium

con-taining 25 mm Hepes at a concentration of 1· 106

cell-sÆmL)1 The macrophage chemotaxis assay was performed

using a polycarbonate membrane with a pore size of 5 lm

(Chemotaxicells; Kurabo, Osaka, Japan) One hundred

microliters of the cell suspension (i.e 105 macrophages)

were added to the upper wells of the Chemotaxicells The

bottom wells were filled with RPMI-1640 medium

contain-ing purified pro-MSP treated or not with HGFA at 37C

for 2 h The recombinant active form of human MSP

(R&D Systems) was used as a positive control After

incu-bation at 37C for 3.5 h, the cells on the upper surface of

the membrane were wiped off with a cotton swab and the

membranes were fixed with 3.7% formaldehyde in NaCl/Pi

and stained with hematoxylin Migration was quantified by

counting the cells on the lower surface in 10 randomly

selected high-power fields (200-fold magnification) To test

the effect of MSP on the morphological changes of

(1· 106cellsÆmL)1) were cultured in serum-free RPMI-1640

medium overnight After incubation, nonadherent cells were

removed and pro-MSP (1.25 nm), pretreated or not with

HGFA, was added to the culture medium After an

addi-tional incubation at 37C for 1 h, morphological changes

of the macrophages were observed by phase-contrast

microscopy

Analysis of molecular forms of MSP in wild-type

and HGFA-deficient mice

The generation of HGFA knockout (HGFA)/)) mice by

the targeting of gene disruption has been reported

previ-ously [18] Sera and EDTA-treated plasma were obtained

from C57BL/6 wild-type (HGFA+/+) and HGFA)/) mice, and diluted 10-fold with phosphate buffer (pH 7.4) Molecular forms of endogenous MSP in the plasma and serum were analyzed by immunoblots To test the effect

of complementation of HGFA activity on serum pro-MSP convertase activity, the diluted serum from an HGFA)/) mouse was incubated with varying concentra-tions of recombinant HGFA at 37C for 2 h, and ana-lyzed by immunoblot For a neutralizing study, the diluted serum from a C57BL/6 mouse was incubated with

or without 400 lgÆmL)1 of HGFA neutralizing anti-body at 37C for the indicated time periods The

immunoblot

Skin injury model

Eight-week-old male wild-type and HGFA)/) mice were deeply anesthetized by intraperitoneal administration of ketamine hydrochloride [100 lgÆ(g body weight))1; Sankyo, Tokyo, Japan] and xylazine [10 lgÆ(g body weight))1; Bayer, Tokyo, Japan] After shaving the dorsal hair and cleaning with 70% ethanol, two full-thickness excisional skin wounds (5 mm in diameter) were made Mice were sac-rificed at 1 or 5 days after the generation of wounds The wounded tissues were excised and used for RT–PCR, immunoblot analysis for pro-MSP processing and routine histological analysis with hematoxylin and eosin staining For control, normal skin tissues were also biopsied (0 day) For RT-PCR, total RNA was prepared with TRIzol (Invi-trogen Japan, Tokyo, Japan) followed by DNase I (Takara Bio, Shiga, Japan) treatment Three micrograms of total RNA were reverse transcribed with a mixture of oligo (dT)12)18 (Invitrogen Japan) and random primers (6-mer) (Takara Bio) using 200 units of ReverTraAce (TOYOBO, Osaka, Japan), and 1/30 of the resultant cDNA was pro-cessed for each PCR with 0.1 lm of both forward and reverse primers and 2.5 units of HotStar Taq DNA poly-merase (Qiagen, Tokyo, Japan) The following primers were used: b-actin: forward, 5¢-TGACAGGATGCAGAAGGA GA; reverse, 5¢-GCTGGAAGGTGGACAGTGAG; CD68: forward, 5¢-TCTACCTGGACTACATGGCGGTGG; reverse, 5¢-ACATGGCCCGAAGTGTCCCTTGTC For immuno-blot, tissues were homogenized on ice in lysis buffer (CelLytic-MT; Sigma-Aldrich) supplemented with prote-ase inhibitor cocktail (Sigma-Aldrich) The extracts were centrifuged at 20 000 g for 20 min at 4C, and the result-ing supernatants were used for immunoblot

Statistical analysis

Statistical analyses were carried out using spss 15.0 (SPSS JAPAN Inc., Tokyo, Japan) P values of less than 0.05 were considered to be statistically significant

Trang 9

This study was supported by a Grant-in-Aid for

Scien-tific Research (B) No 20390114 from the Ministry of

Education, Science, Sports and Culture, Japan We

thank Dr Miyuki Daio for assistance and Dr Takeshi

Shimomura for helpful discussions

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