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Báo cáo khoa học: Regulation of the human leukocyte-derived arginine aminopeptidase/endoplasmic reticulum-aminopeptidase 2 gene by interferon-c pot

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The maximum expression of the gene was achieved by coexpression of IRF-1 and PU.1 in HEK293 cells and IRF-2 suppressed the IRF-1-mediated enhancement of gene expression, suggesting that

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aminopeptidase/endoplasmic reticulum-aminopeptidase 2 gene by interferon-c

Toshihiro Tanioka1,*, Akira Hattori1, Shigehiko Mizutani2 and Masafumi Tsujimoto1

1 Laboratory of Cellular Biochemistry, RIKEN, Wako, Saitama, Japan

2 Department of Obstetrics and Gynecology, Nagoya University School of Medicine, Showa, Nagoya, Japan

Keywords

aminopeptidase; antigen-presentation;

interferon regulatory factor; interferon-c;

PU.1

Correspondence

M Tsujimoto, Laboratory of Cellular

Biochemistry RIKEN (The Institute of

Physical and Chemical Research), 2-1

Hirosawa, Wako-shi, Saitama 351-0198

Japan

Fax: +81 48 462 4670

Tel: +81 48 467 9370

E-mail: tsujimot@riken.jp

*Present address

Laboratory of Medical Information, Showa

University School of Pharmaceutical

Sciences, Shinagawa, Tokyo 142-8555,

Japan

Note

The nucleotide sequence of human

L-RAP(s)1 and L-RAP(s)2 reported in this

paper have been submitted to the

GenBankTM⁄ EMBL ⁄ DDBJ Data Bank with

accession numbers AY028805 and

AB163917, respectively.

(Received 12 October 2004, revised 26

November 2004, accepted 8 December

2004)

doi:10.1111/j.1742-4658.2004.04521.x

The leukocyte-derived arginine aminopeptidase (L-RAP) is the second ami-nopeptidase localized in the endoplasmic reticulum (ER) processing anti-genic peptides presented to major histocompatibility complex (MHC) class

I molecules In this study, the genomic organization of the gene encoding human L-RAP was determined and the regulatory mechanism of its expres-sion was elucidated The entire genomic structure of the L-RAP gene is similar to both placental leucine aminopeptidase (P-LAP) and adipocyte-derived leucine aminopeptidase(A-LAP) genes, confirming the close relation-ship of these three enzymes Interferon (IFN)-c up-regulates the expression

of the L-RAP gene Deletion and site-directed mutagenic analyses of the 5¢-flanking region of the L-RAP gene and electrophoretic mobility shift assay indicated that while interferon regulatory factor (IRF)-2 is important

in the basal condition, IRF-1 is the primary regulator of IFN-c-mediated augmentation of the gene expression In addition, PU.1, a member of the E26 transformation-specific family of transcription factors, also plays a role

in the regulation of gene expression The maximum expression of the gene was achieved by coexpression of IRF-1 and PU.1 in HEK293 cells and IRF-2 suppressed the IRF-1-mediated enhancement of gene expression, suggesting that IFN-c-induced L-RAP gene expression is cooperatively regulated by IRFs and PU.1 transcription factors

Abbreviations

A-LAP, adipocyte-derived leucine aminopeptidase; BAC, bacterial artificial chromosome; CHX, cycloheximide; DTT, dithiothreitol; EMSA, electrophoretic mobility shift assay; ER, endoplasmic reticulum; ERAP, endoplasmic reticulum aminopeptidase; Ets, E26 transformation-specific; IFN, interferon; IL, interleukin; IRF, interferon regulatory factor; L-RAP, leukocyte-derived arginine aminopeptidase; MHC, major histocompatibility complex; P-LAP, placental leucine aminopeptidase; PMSF, phenylmethanesulfonyl fluoride; TAP, transporter associated with antigen processing.

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Aminopeptidases hydrolyze N-terminal amino acids of

proteins or peptide substrates They are distributed

widely in animal and plant tissues as well as in bacteria

and fungi, suggesting that they play important roles in

various biological processes [1] Among them, M1

zinc-metallopeptidases (gluzincins) share the consensus

GAMEN and HEXXH(X)18E zinc-binding motifs

essential for enzymatic activity [2,3] So far, nine

enzymes belonging to the family were identified and

laeverin was recently reported as the tenth member

although its enzymatic activity was not reported [4,5]

In our previous work, we cloned a cDNA for the

pla-cental leucine aminopeptidase (P-LAP)⁄ oxytocinase, a

type II membrane-spanning protein which belongs to

the M1 family of aminopeptidases [6] Subsequently we

cloned cDNAs encoding adipocyte-derived leucine

aminopeptidase (A-LAP) [7], which is also designated as

puromycin-insensitive leucine-specific aminopeptidase

(PILS-AP) or ER-aminopeptidase (ERAP)-1 [8,9], and

leukocyte-derived arginine aminopeptidase (L-RAP)⁄

ERAP2 [4] as highly homologous proteins to P-LAP

Structural and phylogenetic analyses indicated the close

relationship between these three enzymes Therefore we

proposed that they should be classified into the

oxyto-cinase subfamily of M1 aminopeptidases [10]

Recent evidence facilitates new insights into the

biological significance of the oxytocinase subfamily

of aminopeptidases P-LAP⁄ oxytocinase, which is

also designated as insulin-regulated aminopeptidase

(IRAP), was shown to translocate from the

intracellu-lar compartment to the plasma membrane in a

stimu-lus-dependent manner and may regulate concentration

of substrate peptide hormones on the cell surface

[11–14] This enzyme was recently shown to be the

angiotensin IV receptor and may play a role in

memory retention and retrieval [15,16] On the other

hand, we and others reported that A-LAP⁄ ERAP1 is a

final processing enzyme of the precursors of the major

histocompatibility complex (MHC) class I-presented

antigenic peptides [9,17] A-LAP⁄ ERAP1 was also

shown to play roles in blood pressure regulation and

angiogenesis [18–20] Moreover, the enzyme was shown

to bind to cytokine receptors such as tumour necrosis

factor type I receptor, interleukin (IL)-6 a-receptor

and IL-1 type II receptor, and facilitate ectodomain

shedding of these receptors [21–23] As for

L-RAP⁄ ERAP2, we have shown that the enzyme is

retained in the endoplasmic reticulum (ER) and can

trim some precursor peptides to MHC class I ligands

[4] These results indicate that the mammalian

amino-peptidases belonging to the oxytocinase subfamily play

important roles in the regulation of several biological

processes

Precursors of MHC class I-presented peptides with extra N-terminal residues are trimmed to mature epi-topes in the ER [24,25] The peptides are first cleaved from endogenously synthesized proteins by proteasome

or tripeptidyl peptidase II in the cytoplasm, transpor-ted into ER-lumen and then trimmed by certain aminopeptidases Until now, only two aminopeptid-ases (A-LAP⁄ ERAP1 and L-RAP ⁄ ERAP2) have been identified in the ER-lumen and have shown their ability to trim antigenic peptides [4,9,17] Characteris-tically, as in the case of other components included in the presentation of MHC class I ligands such as MHC class I molecules, transporter associated with antigen processing (TAP) and proteasome b-subunits, inter-feron (IFN)-c enhances the expression of these enzymes [24] Although IFN-c-inducible aminopeptid-ases including lens LAP, A-LAP, and L-RAP play roles in the processing or degradation of antigenic peptides [4,9,17,26], the mechanisms of IFN-c-medi-ated regulation of aminopeptidase gene expression have never been examined

In the current study, we have elucidated the genomic structure of human L-RAP gene for the first time Characterization of the promoter region of the gene indicates that while interferon regulatory factor

(IRF)-2 is important in the basal condition, IRF-1 is the primary regulator of IFN-c-mediated augmentation of the gene expression It is also shown that PU.1, a member of the E26 transformation-specific (Ets) family

of transcription factors, plays a role in the regulation

of gene expression Our data provide the molecular basis of regulatory mechanisms of enzymatic activity

of L-RAP, which may play an important role in the processing of antigenic peptides presented to MHC class I molecules in the ER

Results

Genomic organization of human L-RAP gene

To elucidate the genomic organization of the L-RAP gene, we screened a human genome database prepared

in a bacterial artificial chromosome (BAC) This led to the identification of BAC clone RPCI-11-496M2 (accession number AC009126), which contains all exons of the L-RAP gene All exons and intron–exon junctions were determined from the database The sequences of splice junctions obey the GT-AG rule [27] As shown in Fig 1, the gene spans 45 kb and contains 19 exons ranging in size from 28 bp (exon 1)

to 697 bp (exon 2) and the overall structure of the gene is quite similar to both P-LAP and A-LAP genes The first exon includes only the 5¢-untranslated region

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Exon 2 includes the remaining 5¢-untranslated region

and coding sequence for the first 192 amino acids The

GAMEN and HEXXH motifs are encoded in exon 6

and an essential glutamic acid residue located 19

amino acids downstream from the HEXXH motif is

encoded in exon 7 Exon 19 contains the coding

sequence for the last 47 amino acids, stop codon

(TAA) and the 3¢-untranslated region

In our previous work, we obtained a cDNA

enco-ding the truncated form of L-RAP [4] Exon 10 that is

deleted in the P-LAP gene encodes a sequence that

may function as a hinge region [28] As shown in

Fig 1, a transcript encoding this form, termed

L-RAP(s)1 (accession number AY028805), is generated

by differential usage of exon 10 When an intermediate

nucleotide sequence (AACATGgtaag) matching the

GT-AG rule in the exon functions as a splicing donor,

full length L-RAP transcript is generated If the

sequence does not act as a splicing donor, the stop

codon (TGA) in the exon causes the generation of

a truncated form Thereafter, another transcript,

L-RAP(s)2 (accession number AB163917), encoding

the same truncated form was also cloned Differential

usage of exon 15 causes the generation of two

trun-cated forms of the transcripts These results indicate

that at least three mRNAs encoding either the full

length or truncated form of L-RAP protein are

gener-ated from a single gene

Isolation and characterization of human L-RAP

gene promoter

Figure 2 shows the nucleotide sequence of the

5¢-flank-ing region of the human L-RAP gene The

transcrip-tional initiation site of the gene was determined by

5¢ RACE and is shown as nucleotide position +1

in the figure The site (TCAGTC) matches well with

the pyrimidine-rich initiator consensus sequence,

PyPy(A+1)N(T⁄ A)PyPy, in which Py represents a

pyrimidine residue [29] Computer analysis of the

sequence revealed no canonical TATA- or CCAAT-box, suggesting a housekeeping nature for the gene On the other hand, several potential transcription factor binding motifs, such as IRF, GATA-1, Sp-1 and AP-1 were identified in the promoter region of the gene [30]

To characterize the regions regulating transcriptional activity of the L-RAP gene, chimeric reporter plasmids encoding the luciferase gene and different lengths of L-RAP gene were constructed The resultant chimeric constructs were then transfected into HEK293 cells to analyze the promoter activity As a negative control, the promoter-less pGL3 basic plasmid was transfected into the cells As shown in Fig 3A, substantial promo-ter activity was detected when chimeric constructs con-taining the 5¢-flanking sequence upstream from the A

at position )10 were transfected, confirming that the 5¢-flanking sequence of the L-RAP gene is indeed able

to support transcriptional initiation The maximum

Fig 1 Genomic structure of the human L-RAP gene Schematic exon–intron structure of the gene is shown at the center Exons are numbered and depicted as boxes Asterisks indicate the sites used for the generation of different mRNAs described in the text Generation scheme of L-RAP and L-RAP(s) proteins is also shown in the figure Numbers shown in each protein molecule are number of amino acids derived from the respective exons.

Fig 2 Nucleotide sequence of the 5¢-flanking region of the human L-RAP gene The 5¢-flanking region was searched for transcription factor binding sites by TF - SEARCH The exon sequence is shown in uppercase letters, while that of the untranscribed region is given in lowercase letters The transcriptional initiation site shown as +1 was determined by 5¢-RACE For the measurement of promoter activity, the start point of each construct is indicated by an arrowhead.

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promoter activity was obtained with the constructs

containing the sequence from)33 to +5 Deletion of

the sequence from)33 to )17 caused a decrease in the

promoter activity by nearly 50% Further deletion to

T at position )9 caused almost complete loss of

activ-ity Employing Jurkat-T cells, we obtained nearly the

same results

The promoter sequence from)33 to )17 contains a sequence (AGAAAGTGAAAGC) with resemblance to the IRF-E consensus sequence [G(A)AAASYGAA ASY] We next examined the role of this site on the basal promoter activity by constructing mutant plasmid [phLP5(MuI)] from phLP5 plasmid We describe this sequence as the IRF-E site hereafter [31]

Fig 3 Role of the IRF-E site in IFN-c-induced enhancement of L-RAP gene expression in HEK293 cells (A) Luciferase expression plasmids (1 lg) containing sequentially deleted fragments of the L-RAP chimera were transfected into HEK293 cells Luciferase activity was measured

as described in Experimental procedures and normalized to the b-galactosidase activity of a cotransfected internal control plasmid The luci-ferase activity obtained in the basal condition from cells transfected with phLP1 was taken as 100% (B) Enhancing effect of IFN-c is shown

by fold increase in the figure (C) Functional analysis of the IRF-E site of the L-RAP gene promoter in HEK293 cells The phLP5 plasmid hav-ing either an intact or mutated IRF-E site was transfected into HEK293 cells and the luciferase activity was measured as described above Open bars indicate luciferase activity in the basal condition and closed bars in the IFN-c-stimulated condition.

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Although substantial activity was retained, mutation

of the IRF-E site caused a significant decrease in the

promoter activity (Fig 3C) These results suggest that

the IRF-E site is crucial for the maximum promoter

activity of the L-RAP gene in the basal condition

Mechanism of IFN-c-mediated regulation

of L-RAP gene expression

Because L-RAP gene expression is enhanced by IFN-c,

we next examined its regulatory mechanism As an

ini-tial experiment, decay rates of L-RAP mRNAs

pre-pared from Jurkat-T cells treated with or without

IFN-c were compared in the presence of actinomycin

D As shown in Fig 4A, there was little difference

between decay rates of L-RAP mRNAs in the cells

treated with or without IFN-c, indicating that the

cytokine-induced enhancement of mRNA

accumula-tion could not be attributed to the change of stability

of L-RAP mRNA In addition, it was found that the

increase in mRNA accumulation was not observed in

the presence of cycloheximide (CHX), indicating that

de novo protein synthesis was required for the action

of IFN-c (Fig 4B)

To elucidate the role of IFN-c in the regulation of

L-RAP gene expression, the luciferase-reporter assay

was conducted to identify cytokine responsive elements

in the gene As shown in Fig 3A,B, IFN-c induced

about a fourfold increase in the expression of

luci-ferase activity in HEK293 cells transfected with

con-structs containing the sequence from)33 to )17 After

deletion of this sequence, IFN-c had no enhancing

activity These results indicate that the sequence is

essential for the cytokine-induced increase in L-RAP

gene expression

Because the sequence from )33 to )17 contains the

IRF-E site, we next examined the role of this site in

L-RAP gene regulation As shown in Fig 3C,

muta-tion in this site caused complete loss of IFN-c-induced

increase in the luciferase activity, confirming the role

of the IRF-E site in the cytokine-mediated L-RAP

gene regulation These results indicate that the IRF-E

site plays an important role both in the basal and

IFN-c-mediated L-RAP gene expression

On the other hand, it was found that in addition to

the IRF-E site, the Ets site in the promoter also plays

a role in gene expression in Jurkat-T cells As in the

case of HEK293 cells, the IRF-E site was required for

the maximum expression of the gene IFN-c induced

an eightfold increase in the gene expression in cells

transfectied with phLP5 plasmid When Jurkat-T cells

were transfected with plasmids containing the sequence

from )67 to )33 that contains the Ets site, a further

increase (about 13- to 15-fold) was observed (Fig 5A,B) Mutation of either the IRF-E [phLP4(MuI)] or Ets site [phLP4(MuE)] caused a substantial decrease in the IFN-c-mediated gene expression Plasmid having mutations in both sites [phLP4(MuE⁄ MuI)] had little activity, indicating that

in Jurkat-T cells both the IRF and Ets sites are required for maximum enhancement of IFN-c-induced gene expression (Fig 5C) In contrast, it was found that the Ets site had no effect on the promoter activity

in HEK293 (data not shown) We found by RT-PCR

A

B

Fig 4 Requirement of de novo protein synthesis for IFN-c-medi-ated enhancement of L-RAP gene expression (A) Effect of IFN-c

on the stability of L-RAP mRNA Jurkat-T cells were treated with or without 30 ngÆmL)1IFN-c for 12 h at 37 C and further incubated in the presence of 1 l M actinomycin D (ActD) for indicated times After incubation, expression levels of L-RAP mRNAs were meas-ured by Northern blot analysis Densitometric data showing the decay rates of mRNAs are also shown (B) Effect of CHX on the IFN-c-induced enhancement of L-RAP gene expression Jurkat-T cells were preincubated for 3 h in the presence or absence of

1 lgÆmL)1 cycloheximide (CHX) and further incubated with

30 ngÆmL)1IFN-c for 12 h at 37 C L-RAP mRNAs were detected

by Northern blot analysis.

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that expression of PU.1 was detectable in Jurkat-T

cells but not in HEK293 cells (data not shown)

Although the Oct-1 site is located proximal to the

Ets site, mutational analysis indicated that this site

had little effect on the gene expression (data not

shown)

Role of IRFs in the regulation of L-RAP gene expression

Transcription factors, IRF-1 and -2 are known to bind

to the IRF-E site and regulate IFN-c action [31] To examine whether IFN-c affects the expression levels of

Fig 5 Role of the IRF-E and Ets sites in IFN-c-induced enhancement of L-RAP gene expression in Jurkat-T cells (A) Luciferase expression plasmids (10 lg) containing sequentially deleted fragments of the L-RAP chimera were transfected into Jurkat-T cells Cells were then trea-ted with or without 30 ngÆmL)1IFN-c for 15 h at 37 C Luciferase activity was measured as described in Experimental procedures and nor-malized to the b-galactosidase activity of a cotransfected internal control plasmid The luciferase activity obtained in the basal condition from cells transfected with phLP1 was taken as 100% (B) Enhancing effect of IFN-c is shown by fold increase in the figure (C) Mutational analy-sis of the IRF-E and Ets sites of the L-RAP gene promoter in Jurkat-T cells The phLP4 plasmid having either intact or mutated sites shown

in the left panel of the figure was transfected into Jurkat-T cells and luciferase activity was measured Open bars indicate luciferase activity

in the basal condition and closed bars in the IFN-c-stimulated condition.

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IRF-1 and -2 proteins, HEK293 cells were cultured in

the presence or absence of the cytokine (Fig 6A)

When compared with untreated cells, an increase in the

expression level of IRF-1 was observed in cells treated

with IFN-c, indicating that IRF-1 is a candidate for the

mediator of IFN-c action In contrast, no effect was

observed on the expression of IRF-2 protein

To determine whether IRF-1 and -2 bind to the

IRF-E site in the L-RAP promoter sequence, we

per-formed an electrophoretic mobility shift assay (EMSA)

using nuclear extracts from HEK293 cells treated with

or without IFN-c (Fig 6B) An oligonucleotide probe corresponding to the IRF-E site bound to nuclear extracts prepared from both IFN-c-treated and untreated cells In spite of the presence of an unex-pected nonspecific large band, bands shown as IRF-1 and -2 in Fig 6 were replaced by unlabeled oligo-nucleotide competitor, indicating their specificity The specificity of these bands was also confirmed by oligo-nucleotide with mutations at the IRF-E site Although repeated trials were made, we could not remove the nonspecific bands

To identify transcription factors bound to the

IRF-E site, supershift assay was performed using antibodies raised against IRF-1 and IRF-2 When anti-(IRF-1) IgG was employed, supershift was observed only in the IFN-c-treated cells On the other hand, anti-(IRF-2) IgG caused supershift in both treated and untreated cells Moreover, appearance of two supershift bands and complete disappearance of the oligonucleotide spe-cific bands was observed in the presence of both anti-bodies These results suggest that while in untreated cells IRF-2 but not IRF-1 was bound to the IRF-E site, IRF-1 and IRF-2 co-occupied the site after treat-ment with IFN-c We obtained the same results in Jur-kat-T cells However, it should be noted here that an IFN-c-inducible band other than IRF-1 and -2 was also observed in this experiment, suggesting that tran-scription factors other than IRF-1 and -2 also partici-pate in the regulation of L-RAP gene expression

A

B

Fig 6 Role of IRF-1 and PU.1 in the expression of the L-RAP gene (A) IFN-c-induced increase in the expression of IRF-1 protein in HEK293 cells HEK293 cells were treated with or without

30 ngÆmL)1IFN-c for 5 h at 37 C IRF-1 and IRF-2 in the nuclear extracts were measured by Western blot analysis (B) Binding of IRF-1 and IRF-2 to the IRF-E site of the L-RAP gene HEK293 cells were treated with or without 30 ngÆmL)1 IFN-c for 5 h at 37 C EMSAs were then performed in nuclear extracts using a probe con-taining the IRF-E site from the L-RAP gene promoter Supershifts were conducted using antibodies against indicated factors Arrow-heads indicate the positions of IRF-1 and IRF-2 Arrow indicates the position of unidentified IFN-c-inducible IRF-E binding protein Lanes shown as probe are loaded only with labeled probe (C) Enhance-ment of promoter activity of the L-RAP gene by transcription fac-tors HEK293 cells were cotransfected with phLP4 (0.5 lg) and various expression plasmids (0.5 lg) shown in the figure After

24 h, luciferase activity was measured as described in Experimental procedures Enhancing effects of the transcription factors are shown by fold increase using cells transfected only with phLP4 or

as a control (D) Induction of endogenous L-RAP gene expression

by transcription factors HEK293 cells were transfected with expression plasmids (0.5 lg) shown in the figure For RT-PCR ana-lysis, total RNA was extracted after 24 h-incubation Relative expression level is shown by fold increase in the figure using mock-transfected cell as a control.

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To further elucidate the role of IRF-1 and -2 in the

regulation of L-RAP gene expression, phLP4 plasmid

was cotransfected either with pTARGET-IRF-1 or

pTARGET-IRF-2 plasmid into HEK293 cells

(Fig 6C) IRF-1 overexpressed in HEK293 cells

induced 8.6-fold increase in the luciferase activity,

indi-cating that IRF-1 is indeed able to augment L-RAP

gene expression IRF-2 and PU.1, a transcription

fac-tor which is known to bind to the Ets site [32], also

had enhancing effects on the luciferase activity, and a

4.7- and 2.1-fold increase in the gene expression was

observed, respectively Furthermore, maximum gene

expression (18.3-fold increase) was achieved in a

syner-gistic manner when IRF-1 and PU.1 were coexpressed

On the other hand, coexpression of IRF-1 and IRF-2

caused lower expression of luciferase activity than

expected, suggesting that IRF-2 suppressed

IRF-1-mediated augmentation of the gene expression We

also examined other Ets site-binding factors such as

Ets-1 and Ets-2 and found that they had little effect

on the gene expression (data not shown) When the

same experiments were performed using either

phLP4(MuE), phLP4(MuI) or phLP4(MuE⁄ MuI), no

synergistic effect between IRF-1 and PU.1 was

observed (data not shown), confirming that native sites

of both IRF-E and PU.1 are required for the

synergis-tic action Taken together these results indicate that

while IRF-2 plays a role in the basal condition, IRF-1

can mediate IFN-c-stimulated L-RAP gene expression

synergistically with PU.1

To determine whether IRF-1 indeed mediates

L-RAP gene expression in vivo, we next examined the

effect of transcription factors on the expression of

endogenous L-RAP gene Several combinations of

plasmids were transfected into HEK293 cells and their

ability to enhance gene expression was examined by

RT-PCR (Fig 6D) In mock-transfected cells, L-RAP

mRNA was barely detectable Among trancription

fac-tors tested, the highest expression was achieved when

IRF-1 was overexpressed Further increase in the gene

expression was observed when IRF-1 and PU.1 were

coexpressed In contrast, IRF-2 suppressed

IRF-1-mediated increase in the gene expression, although

IRF-2 alone had some enhancing activity These

results further confirm the roles of transcription factors

in the expression of the L-RAP gene

Discussion

L-RAP⁄ ERAP2 is a newly identified ER

aminopepti-dase and the third member of the oxytocinase

sub-family of M1 sub-family of aminopeptidases [4] It was

suggested by its ability to generate certain antigenic

peptides that the enzyme plays an important role in the processing of antigenic peptides presented to MHC class I molecules in the ER In this study, we deter-mined the genomic structure of the human L-RAP gene and characterized the regulatory mechanisms of the gene expression It was found that two forms of the L-RAP proteins are generated by alternative spli-cing While the full length form exerts distinct amino-peptidase activity, the truncated form has no enzymatic activity [4] It is necessary to elucidate the relationship between these two forms

The entire genomic structure of the L-RAP gene is quite similar to the P-LAP and A-LAP genes [28,33] The GAMEN and HEXXH(X)18E motifs essential for the enzymatic activity are encoded by exons 6 and 7

of the respective genes Becasue these three genes are located contiguously around human chromosome 5q15, between versican and calpastatin [28], these data further confirm the latest divergence of the genes from

a common ancestral gene

Expression of L-RAP is up-regulated by IFN-c [4] IFN-c also stimulates the induction of components included in the processing of MHC class-I ligands [24] Considering the evidence that suggest a role of L-RAP

as an antigen-trimming enzyme in the ER, it is import-ant to elucidate the mechanism of the IFN-c-induced increase in L-RAP gene expression The transient expression of the 5¢-flanking region of the L-RAP gene fused to the luciferase gene in either HEK293 or Jurkat-T cells allowed the analysis of basal and IFN-c-mediated regulation of promoter activity We demon-strated by deletion analysis that the sequence ranging from )16 to )10 carries the minimum promoter activ-ity of the gene In addition, mutational analysis sug-gested that the IRF-E site is required for the maximum enhancement of gene expression in the basal condition Because EMSA indicated that IRF-2 is associated with the IRF-E site in unstimulated HEK293 cells, our data suggest that IRF-2 can aug-ment the transcription of the L-RAP gene in the basal condition In fact, IRF-2 had some enhancing effect

on the gene expression when overexpressed in HEK293 cells Although it is generally considered that IRF-2 is

a negative regulator of gene expression [31], it has also been shown to up-regulate the expression of several genes such as histone 4 and VCAM-1 [34,35] However,

we could not completely rule out the possible contribu-tion of other IRF-E site-binding proteins [31], because

an IFN-c-inducible IRF-E binding protein other than IRF-1 and IRF-2 was detectable by EMSA, even in the basal condition Identification and elucidation of the role of this protein in L-RAP gene expression is required

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It is worthy of noting here that both P-LAP and

A-LAP promoters also contain IRF-E sites [28,33] It

was reported that IFN-c had no enhancing effect on

P-LAPgene expression [33] In our preliminary results,

the IRF-E site in the A-LAP promoter had little effect

on IFN-c-induced gene expression, raising the

possib-lity that IFN-c affects the expression of the A-LAP

gene differently from that of the L-RAP gene

IFN-c-induced increase in L-RAP gene expression

required de novo protein synthesis and it was found

that IRF-1 was induced by IFN-c Deletion and

muta-tional analyses indicated that the IRF-E site is

res-ponsible for the cytokine-mediated increase in gene

expression in HEK293 cells Mutation of the site

caused loss of the cytokine action EMSA indicated

that while IRF-2 but not IRF-1 bound to the IRF-E

site in the basal condition, binding of both IRF-1 and

-2 were detectable after treatment with IFN-c

More-over, transfection of the IRF-1 expression plasmid

into HEK293 cells caused an increase in the gene

expression These results strongly suggest that IRF-1 is

a primary mediator of IFN-c-mediated enhancement

of L-RAP gene expression in HEK293 cells Because

coexpression of IRF-1 and IRF-2 caused a decrease in

L-RAP gene expression, it is plausible that IRF-2 acts

as a negative regulator of IRF-1-mediated

enhance-ment of gene expression, by co-occupation of the

IRF-E site with IRF-1 in IFN-c-treated cells

On the other hand, the mechanism of IFN-c-induced

L-RAPgene regulation in Jurkat-T cells is rather

com-plex It is obvious that in addition to the IRF-E site,

the Ets site located between )63 and )56 also plays a

role in the maximum enhancement of IFN-c-induced

gene expression It is unlikely that another Ets site

located between )352 and )345 plays a role in the

gene expression, because deletion of this sequence had

little effect on the enhancement of the gene expression

Among the Ets family transcription factors tested, only

PU.1 could mediate the cytokine action The

expres-sion of PU.1 is limited to hematopoietic lineages and

is necessary for the differentiation of myeloid cell

line-ages such as macrophline-ages and osteoclasts [36,37] Our

analysis by RT-PCR indicated the expression of PU.1

in Jurkat-T cells but not HEK293 cells Therefore, it is

plausible that the lineage-dependent expression of

PU.1 may determine the expression level of the L-RAP

gene Consistent with this notion, IRF-1 and PU.1

mediated the maximum expression of both reporter

and endogenous genes when overexpressed, even in

HEK293 cells

The molecular basis of the cooperative action of

transcription factors is considered to be the physical

interaction of the transcription factors involved [37]

For instance, it was reported that IRFs and PU.1 synergistically mediate the transcriptional enhancement

of human interleukin-1 (IL-1)b gene expression It was

suggested by physical interaction analysis that the fac-tors might function together as an enhanceosome [38]

As shown in other genes [39,40], it is plausible that IRF-1 mediates the IFN-c-stimulated L-RAP gene expression through interaction with PU.1 On the other hand, it is possible that IRF-2 acts independently from PU.1 to enhance the gene expression in the basal con-dition Because binding of IRF-2 to the IRF-E site was still observed after IFN-c treatment, it is possible to speculate that IRF-2 modulates the gene expression by interacting with IRF-1 in the IRF-E site of the L-RAP gene As with the L-RAP gene, Gobin et al reported that binding of IRF-2 to the MHC class I molecule promoter was observed after IFN-c treatment [41] It was also reported that IRF-2 co-occupies the IRF-E site of the class II transactivator type IV promoter with IRF-1 and synergistically activates the promoter [42] L-RAP⁄ ERAP2 is the second ER-lumenal amino-peptidase to be determined, and can trim certain pre-cursors of antigenic peptides presented to MHC class I molecules [4], suggesting the potential significance of this enzyme in antigen processing In this study, we characterized the L-RAP gene for the first time We have shown that the IRF-E site located in the proximal region of the transcription initiation site plays a pivotal role in the regulation of human L-RAP gene expres-sion both in the basal and IFN-c-stimulated condi-tions As shown in several genes [43–45], it was found that while IRF-2 plays a role in the basal condition, IRF-1 is the primary regulator of the gene expression induced by IFN-c However, transcription factor(s) other than IRF-1 and -2 may also regulate L-RAP gene expression Further works are required to exam-ine the involvement of other transcription factors that bind to the IRF-E site Considering the significance of the processing of antigen presented to MHC class I molecules in several pathophysiological conditions such

as virus infection, tumor generation and self-antigen generation, it is important to elucidate the total aspects

of the antigen presentation process including the regu-latory mechanisms of L-RAP gene expression

Experimental procedures

Identification of the human L-RAP gene

A genomic sequence of  178 kb from the Gen-BankTM⁄ EMBL ⁄ DDBJ Data Bank was obtained This sequence encompasses a region on human chromosome 5q15 where the known P-LAP gene is located The

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loca-tions of the exons of the L-RAP gene were determined

using the blast program

Cell culture

Jurkat-T cells were obtained from the RIKEN Cell Bank

(Tsukuba, Japan) and maintained in RPMI 1640 Human

embryonic kidney (HEK) 293 cells were purchased from

ATCC (Manassas, VA, USA) and maintained in RPMI

1640 All media used in this study was from Sigma (St

Louis, MO, USA) and supplemented with 10% fetal

bovine serum (JRH Biosciences, Lenexa, KS, USA),

peni-cillin G, and streptomycin (Meiji Seika Co., Kanagawa,

Japan) The cells were cultured in a humidified 5% CO2

and 95% air incubator at 37C

Analysis of promoter activity

The human L-RAP 5¢-flanking region and its fragments,

which include the transcriptional initiation site, were

amplified by PCR from a BAC clone PCR products

were inserted into the promoter-less plasmid, pGL3 basic

(Promega, Madison, WI, USA) The nucleotide sequence

of the primers used for PCR amplification were as

follows (5¢-3¢): CGGGTACCTGAACCAGCTAGTACT

TACTG (sense strand of the sequence from )88 to )68)

for phLP3; CGGGTACCTACTCAGGAAGCATGC

AAGT (sense strand of the sequence from )67 to )47)

for phLP4; CGGGTACCACAGAAAGTGAAAGCA

(sense strand of the sequence from )33 to )18) for

phLP5; CGACGCGTTGACTGAAGGGGAATTTACTTT

(antisense strand of the sequence from )17 to +5) for

all constructs For phLP6 and 7, plasmids were

construc-ted by using synthetic oligonucleotides corresponding to

the 5¢-flanking region We also constructed phLP1 and

2 plasmids by sequetial deletion of SmaI and Van91I

fragment from phLP0 covering the sequence from )1042

to +5

Mutagenesis of the IRF and Ets sites was performed

by using mutated oligonucleotides when constructing

the respective plasmids For making mutations in IRF,

5¢-CACAGAGGGTGAGGGCAAAAGTAAATTCCCCTT

CAGTCAA-3¢ (sense sequence of mutant IRF-E site) and

5¢-CGCGTTGACTGAAGGGGAATTTACTTTTGCCCT

CACCCTCTGTGGTAC-3¢ (antisense sequence of mutant

IRF-E site) were used For the Ets mutant, 5¢-GGGGTA

CCTACTCAAAGAGCATGCAAAGT-3¢ (sense sequence

of mutant Ets site) and 5¢-CGACGCGTTGACTGAAGG

GGAATTTACTTT-3¢ (antisense sequence of mutant Ets

site) were used For the construction of the double mutant

plasmid, 5¢-GGGGTACCTACTCAAAGAGCATGCAAA

GT-3¢ and 5¢-CGACGCGTTGACTGAAGGGGAATTTA

CTTTTGCCCTCACCCTCTGTTCTAA-3¢ (antisense

sequ-ence of mutant IRF-E site) were used The mutated

nucleo-tide sequences are underlined

The PU.1 expression plasmid pcDNA3-PU.1 which was constructed by using pcDNA3 (Invitrogen, Carlsbad, CA, USA) was obtained from M Matsumoto of Saitama Med-ical School (Saitama, Japan) [46]

Transfection and luciferase assay For transfection of the reporter plasmid, HEK293 cells were plated on 24 well plates at a density of 1· 105 cells per well on the day before transfection, while Jurkat-T cells (1· 106 cells) were plated in a 100 mm dish Plasmid DNA was mixed with LipofectAmine (Invitorogen) and transfected into HEK293 cells following the manufacturer’s protocol A pCMVb (0.5 lg) plasmid was employed as an internal control of transfection efficiency To transfect into Jurkat-T cells, the electroporation (225 V, 350 lF) method was employed, using Electro Cell Manipulator (BTX, San Diego, CA, USA) After 24 h of transfection, the cells were washed three times with NaCl⁄ Piand then lysed in reporter lysis buffer (Promega) The luciferase activity was then measured with a luciferase assay system (Promega) accord-ing to the manufacturer’s instruction Luciferase activity was measured in triplicate, averaged, and then normalized

to b-galactosidase activity to correct for transfection effi-ciency b-Galactosidase activity was measured using o-nitro-phenyl-b-d-galactopyranoside as a substrate

Electrophoretic mobility shift assay Double-stranded oligonucleotides containing the consensus sequence IRF-E site were radiolabeled with [32P]dCTP[aP]

at the 3¢ end with a Klenow fragment and then purified with a MicroSpin column (Amersham Biosciences, Piscata-way, NJ, USA) The sense sequences of the synthesized oligonucleotides used were as follows: for the native IRF-E site (GAACAGAAAGTGAAAG) in the human L-RAP 5¢-flanking region, and for mutation in the IRF-E site (GAACAGAGGGTGAGGG) and the antisense sequences

of synthesized oligonucleotides used were as follows: for native IRF-E site (TTTGCTTTCACTTTCT) in the human L-RAP 5¢-flanking region, for mutation in IRF-E site (TTTGCCCTCACCCTCT) The mutated nucleotide sequ-ences are underlined

Nuclear extracts of the cells were prepared as follows: the cells suspended in 500 lL of ice-cold buffer A [10 mm He-pes, 10 mm KCl, 1.5 mm MgCl2, 0.5 mm dithiothreitol (DTT), 0.2 mm phenylmethanesulfonyl fluoride (PMSF):

pH 7.9] were lysed in a Dounce-homogenizer and then the nuclei were pelleted by centrifugation at 3300 g for 15 min The pellets were then suspended in an equal volume of low salt buffer (20 mm Hepes, 20 mm KCl, 1.5 mm MgCl2, 0.2 mm EDTA, 0.5 mm DTT, 0.2 mm PMSF, 25% glycerol:

pH 7.9) Thereafter, half volume of high salt buffer (20 mm Hepes, 1.4 m KCl, 1.5 mm MgCl2, 0.2 mm EDTA, 0.5 mm DTT, 0.2 mm PMSF, 25% glycerol: pH 7.9) was added

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