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In Drosophila SL2 cells, which lack the Sp family of tran-scription factors, forced expression of Sp1 or Sp3 enhanced transcription from the Dnmt1 promoter.. To understand the regulation

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Regulation of transcription of the Dnmt1 gene by Sp1 and Sp3 zinc finger proteins

Shotaro Kishikawa1,2, Takehide Murata1, Hiromichi Kimura2, Kunio Shiota2and Kazunari K Yokoyama1 1

Gene Engineering Division, Department of Biological Systems, BioResource Center, RIKEN (The Institute of Physical & Chemical Research), Japan;2Department of Animal Resource Science/Veterinary Medical Sciences, The University of Tokyo, Japan

The Sp family is a family of transcription factors that

bind to cis-elements in the promoter regions of various

genes Regulation of transcription by Sp proteins is based

on interactions between a GC-rich binding site

(GGGCGG)in DNA and C-terminal zinc finger motifs

in the proteins In this study, we characterized the

GC-rich promoter of the gene for the DNA

methyl-transferase (Dnmt1)that is responsible for methylation of

cytosine residues in mammals and plays a role in gene

silencing We found that a cis-element (nucleotides )161

to )147)was essential for the expression of the mouse

gene for Dnmt1 DNA-binding assays indicated that

transcription factors Sp1 and Sp3 bound to the same

cis-element in this region in a dose-dependent manner In

Drosophila SL2 cells, which lack the Sp family of tran-scription factors, forced expression of Sp1 or Sp3 enhanced transcription from the Dnmt1 promoter Sti-mulation by Sp1 and Sp3 were independent phenomena Furthermore, cotransfection reporter assays with a p300-expression plasmid revealed the activation of the promoter of the Dnmt1 gene in the presence of Sp3 The transcriptional coactivator p300 interacted with Sp3 in vivo and in vitro Our results indicate that expression of the Dnmt1 gene is controled by Sp1 and Sp3 and that p300 is involved in the activation by Sp3

Keywords: Dnmt1 gene; activation of transcription; Sp1; Sp3; p300

Transcription is regulated by the combinational actions of

proteins that bind to distinct promoter and enhancer

elements In general, a limited number of cis-acting

DNA elements is recognized, not that by a single

transcrip-tion factor exclusively but, rather, by a set of different

proteins that are structurally related [1] The promoter

regions of many eukaryotic genes contain GC-rich sequences

[2] and some of the most widely distributed promoter

elements are GC boxes and related motifs [2]

The Sp family of transcription factors includes the

proteins Sp1, Sp3 and Sp4, which recognize and bind to

GC boxes as well as to GT/A-rich motifs with similar

affinity, and Sp2, which binds preferentially to GT/A-rich

sequences [2,3] Sp1 and Sp3 are expressed in a wide variety

of mammalian cells whereas Sp4 has been detected

predominantly in neuronal tissues The regulation of gene

expression by Sp transcription factors is complex Although

certain promoters can be activated by either Sp1 or Sp3 in

assays in vivo and are occasionally activated by both Sp1

and Sp3 that act in a synergistic manner [4–6], there are

other promoters that show a definite preference for Sp1 or

Sp3 [7] Furthermore, Sp3 can function as an activator or a

repressor of transcription, depending on the gene in question [8,9]

The genes for several mammalian activators and repressors of transcription have been cloned The gene for p300 was first cloned as the gene for an E1A-associated protein with properties of a transcriptional adapter [10] The protein was found later to possess intrinsic histone acetyltransferase (HAT)activity and

to function as a coactivator in MyoD-, p53-, and SRC-1-mediated transcription [11,12] Furthermore, p300 appears to play a critical role in progression of the cell cycle and the differentiation of cells [11,12]

The methylation of DNA plays a role in the regulation

of gene expression [13,14], genomic imprinting [15] and inactivation of the X chromosome [16] and it has been shown to be essential for mammalian development [17,18] Altered patterns of DNA methylation has been implicated

in tumorigenesis [19] However, the mechanisms by which DNA methylation is regulated during development and tumorigenesis remain largely unknown Five distinct families of gene for DNA methyltransferases, designated Dnmt1, Dnmt2, Dnmt3a, Dnmt3b and Dnmt3L, have been identified in mammalian cells [20] Dnmt1 is expressed constitutively in proliferating cells, it is associated with foci

of DNA replication [21] and methylates CpG dinucleo-tides [22] These findings are consistent with the hypothesis that Dnmt1 is a maintenance methyltransferase that restores appropriate patterns of DNA methylation to the genome shortly after DNA replication [23] Representative sites for initiation of transcription have been found in the promoter of the Dnmt1 gene, namely, an oocyte-specific site, a somatic cell-specific site and a spermatocyte-specific site In adult somatic cells, most of the available data indicate that the identification by Bigey et al of many

Correspondence to K K Yokoyama, Gene Engineering Division,

Department of Biological Systems, BioResource Center, Tsukuba

Institute, RIKEN (The Institute of Physical & Chemical Research),

3-1-1 Koyadai, Tsukuba, Ibaraki 305-0074, Japan.

Fax: + 81 298 36 9120, Tel.: + 81 298 36 3612,

E-mail: kazu@rtc.riken.go.jp

Abbreviations: ChIP, chromatin immunoprecipitate; ODN,

oligodeoxynucleotides; HAT, histone acetyltransferase.

(Received 28 January 2002, revised 25 April 2002,

accepted 30 April 2002)

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sites of initiation of transcription of Dnmt1 [24] might

have been a mistake and that there is a single site only

[23]

To understand the regulation of expression of the

Dnmt1 gene in somatic cells, it is necessary to identify

and characterize the promoter region of this gene Previous studies showed that the 5¢ end region of the Dnmt1 gene is typical of a TATA-less and GC-rich promoter [24] However, specific cis- or trans-acting elements involved in the regulation of that promoter remain to be identified

In this study, we identified a cis-element located between nucleotides)161 and )147 that appeared to be activated independently by Sp1 and Sp3 Moreover, the p300 coactivator appeared to be involved in the Sp3-mediated activation of the mouse Dnmt1 promoter in somatic cells

M A T E R I A L S A N D M E T H O D S

Cells, plasmids and materials Mouse NIH3T3 cells and Drosophila SL2 cells were obtained from the JCRB Cell Bank (Tokyo, Japan)and from S Kojima at RIKEN (Tsukuba, Japan) pCMV-Sp1 and pGEX-Sp1 were provided by R Chiu of the UCLA School of Medicine (Los Angeles, CA, USA) Plasmids pPac, pPacSp1, pPacUSp3, pGEX-Sp3 and pCMV-Sp3 were gifts from G Suske at Philipps-Universita¨t (Mar-burg, Germany) GST–p300 (amino acids 1–596), GST– p300 (amino acids 744–1571)and GST–p300 (amino acids 1,572–2414)were obtained from Y Shi of Harvard Medical School (Boston, MA, USA) Plasmid pCi-p300 was a gift form Y Nakatani of the Dana Farber Cancer Research Institute (Boston, MA, USA) DNA fragments

of the mouse Dnmt1 promoter were excised with appro-priate restriction enzymes (D1 to D5 in Fig 1A) Each DNA fragment was inserted into the Nhe1 and Xho1 sites

of pGL3-basic (Promega Co., Madison, WI, USA) The integrating of all of the above recombinant plasmids was verified by sequence analysis Antibodies against Sp1 (PEP2), Sp3 (D-20), AP-2 (C-18)and p300 (C-20)were purchased from Santa Cruz Biotechnology Inc (Santa Cruz, CA, USA)

Fig 1 Characterization of the promoter of the mouse Dnmt1 gene (A) Transcriptional activity of the mouse Dnmt1 promoter in NIH3T3 cells The top diagram shows the mouse Dnmt1 gene and the lower diagram shows the variously deleted promoters fused to a gene for luciferase (gray rectangle) The corresponding luciferase activities are also shown Numbering is relative to the first site of initiation of transcription (+1) Reporter plasmid D5 has the deletion of nucleo-tides )173 to )120 of D2 fusion construct NIH3T3 cells were trans-fected with plasmids that encoded the various constructs and luciferase activity was measured as described in the text Promoter activities are expressed relative to the activity associated with reporter plasmid D4, which was taken arbitrarily as 1.0 All values are the averages of results from at least three experiments and the standard deviation for each value is indicated (B)Nucleotide sequence of the 5¢ flanking region of the mouse gene for Dnmt1 Bases are numbered relative to the site

of initiation of transcription (+1) P1, P2 and P3 indicate the probes used for EMSAs (C)Binding of transcription factors to the Dnmt1 promoter Binding of transcription factors to the region between nucleotides )173 and )117 of the Dnmt1 promoter was analyzed by EMSAs with 32 P-labeled probes P1, P2, and P3 (1 · 10 4 c.p.m)and

5 lg protein of nuclear extract (NE)from NIH3T3 cells Three shifted protein-DNA complexes are indicated by arrows (B1–B3).

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Site-directed mutagenesis

Site-directed mutagenesis was performed with a

Quick-Change Site-Directed Mutagenesis Kit (Stratagene,

La Jolla, CA, USA)using various oligodeoxynucleotide

primers (n1 to n10 in Fig 2D)and a fragment of the

Dnmt1 promoter (nucleotides)220 to +79)as template

The mutated DNA fragments were subcloned into the

Nhe1 and Xho1 sites of pGL3-basic (Promega Co.) The

integrity of all of the vectors was verified by sequence

analysis

Recombinant proteins

Glutathione S-transferase (GST), GST–Sp1, GST–Sp3,

GST–p300 (amino acids 1–596), GST–p300 (amino acids

744–1571)and p300 (amino acids 1572–2414)were prepared

as described previously [25].35S-Labeled Sp1 and Sp3 were

synthesized in a TNT Reticulocyte Lysate System (Promega

Co.), according to the manufacturer’s protocol

Cell culture, transfections and assays of promoter

activity

Mouse NIH3T3 and F9 cells and human HeLa and 293

cells were grown in Dulbecco’s modified Eagle’s medium

(Nissui Pharmaceutical Co., Tokyo, Japan)with 10% fetal

bovine serum (Invitrogen BV, Groningen, the Netherlands)

at 37C in a humidified atmosphere of 5% CO2 in air

Schneider’s Drosophila SL2 cells were maintained in Shields

and Sang M3 insect medium (Sigma–Aldrich Japan Co.,

Tokyo, Japan)supplemented with 10% fetal bovine serum

at 22C in air One day prior to transfection, mammalian

cells were seeded in 12-well plates at 8· 104cells per well

and SL2 cells were seeded at 1· 106cells per well Cells

were transfected with the indicated amount of reporter

plasmid using LipofectamineTM2000 (Invitrogen BV) Cell

extracts were prepared 24 or 48 h after transfection

Promoter activity was determined with a Luciferase Assay

System (Promega Co.)as described by the manufacturer

Electrophoretic mobility shift assays (EMSAs)

Nuclear extracts were prepared from NIH3T3, HeLa, 293

and F9 cells and EMSAs were performed as described

previously [26] DNA probes were radiolabeled with T4 polynucleotide kinase (New England BioLabs Inc., Beverly,

MA, USA)and [c-32P]ATP (Amersham Pharmacia

Fig 2 Identification of a cis-element in the Dnmt1 promoter (A)

EMSAs were performed with mutated oligodeoxynucleotides as

competitors and the wild-type ODN probe (B)Oligodeoxynucleotides

used as competitors are listed Wt, Sequence of the wild-type probes.

M1 through M11, the GAATTC sequence was introduced into the

wild-type motif to generate the mutant sequences (C)Assays of

luciferase reporter activity in NIH3T3 cells using mutated reporter

constructs NIH3T3 cells were transfected with the reporter plasmid

D2 or with plasmids that included the sequences n1 through n10 and

luciferase activities were measured, and compared with that obtained

with NIH3T3 cells that had been transfected with reporter plasmid D4

(500 ng) These assays were repeated at least three times and the

standard deviation for each average value is indicated (D)Nucleotide

sequences obtained by site-directed mutagenesis Wt, wild-type

sequence; n1 through n10, TT di-nucleotides were introduced, as

indicated, to generate the mutants.

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Biotech., Uppsala, Sweden) The binding reaction was

performed in 20 lL of buffer that contained 25 mM

N-2-Hydroxyethylpiperazine-N¢-2-ethanesulfonic acid/KOH

(Hepes/KOH, pH 7.9), 25 mMKCl, 5 mMMgCl2, 50 mM

ZnSO4, 1 lg poly(dI-dC), and a nuclear extract or purified

GST-fusion protein In some cases, competitors or

anti-bodies were added to reaction mixtures which were then

incubated on ice for 20 min After addition of the

32P-labeled DNA probe, the mixture was incubated for a

further 20 min on ice Products of reactions were resolved

on a 5% polyacrylamide gel in 0.5· Tris/borate/EDTA

buffer Electrophoresis was performed at 180 V for 3 h at

4C

Immunoprecipitation of chromatin (ChIP)

NIH3T3 cells were fixed in 1% formaldehyde at room

temperature for 15 min Chromatin was prepared with a

kit from Upstate Biotechnology (Lake Placid, NY, USA)

according to the recommendations of the manufacturer,

with eight 10-s pulses of sonication at 10-s intervals, which

yielded chromatin fragments of  1.0-kb in length

Equivalent amounts of chromatin were

immunoprecipi-tated with the indicated antibody or with irrelevant IgG,

as a control, at 4C for 5 h Immunocomplexes were then

recovered by addition of protein A/G PLUS-agarose

beads (Santa Cruz Biotech., Santa Cruz, CA, USA)with

incubation at 4C for 2 h After the beads had been

washed extensively, DNA was eluted and cross-linking

was reversed by addition of 200 lL of elution buffer (1%

SDS/0.1M NaHCO3)and incubated overnight at 65C

DNA was extracted with phenol/chloroform (1 : 1, v/v),

precipitated in ethanol and then analyzed by PCR with

primers that corresponded to the cis-element (nucleotides

)220 to +79), namely, 5¢-AAGGCTAGCCAGAGTCA

TCCTCTGC-3¢ (forward direction)and 5¢-GCGCTCG

AGCTTGCAGGTTGCAGAC-3¢ (reverse direction) PCR

was performed for 35 cycles and products were analyzed

by agarose gel electrophoresis

Immunoprecipitation and Western blotting

Cell pellets were lysed in RIPA buffer (1· NaCl/Pi,

1% Nonidet P-40, 0.5% sodium deoxycholate, 0.1%

SDS, 100 lgÆmL)1phenylmethanesulfonyl fluoride, 1 mM

sodium orthovanadate and 2 lgÆmL)1aprotinin]

Whole-cell extracts (500 lg proteins)were incubated with 2 lg of

antibody [Sp1 Ig, Sp3 Ig, (AP-2)Ig,

anti-p300 Ig or nonimmunized rabbit IgG] for 1 h, and then

30 lL of a suspension of protein A/G PLUS-agarose

beads (Santa Cruz)were added After incubation for 1 h

at 4C, immunoprecipitates were gently washed three

times with NaCl/Pi, boiled and subjected to SDS/PAGE

(7% acrylamide gel) Proteins were electroblotted onto a

poly(vinylidene difluoride)membrane filter and blocked

for 1 h in Blotto A [10 mM Tris/HCl (pH 8.0), 150 mM

NaCl, 5% skim milk, 0.05% Tween-20], and then

incubated with 2 lgÆmL)1of primary antibody (anti-Sp1

Ig or anti-Sp3 Ig)in BlottoA for 1 h Finally, 30 lL of a

solution of horseradish peroxidase conjugated-secondary

antibody (New England Biolabs)in Blotto A were added

Antibody–HRP complexes were detected with ECL

West-ern blotting detection reagents (Amersham Pharmacia

Biotech.)according to the instructions from the manufac-turer

‘GST-pull down’ assay Two micrograms of GST–protein and 35S-labeled Sp3 (3· 103c.p.m)that had been in vitro translated in a final volume of 500 lL of binding buffer [250 mMNaCl, 50 mM Hepes/KOH (pH 7.5), 0.5 mMEDTA, 0.1% Nonidet P-40, 0.2 mMphenylmethanesulfonyl fluoride, 1 mM dithiothre-itol and 500 lgÆmL)1BSA], were incubated at 4C for 1 h and then 30 lL of a suspension of glutathione–Sepharose 4B (Amersham Pharmacia Biotech)were added After incubation for 1 h at 4C, samples were gently washed three times with NaCl/Pi, boiled and fractionated by SDS/ PAGE (7% acrylamide gel)

R E S U L T S

Identification ofcis-elements that control expression of the Dnmt1 gene

We subcloned a 2.0-kb DNA fragment that contained the Dnmt1 promoter region from a mouse genomic clone into pGL3-basic, a vector that includes a gene for luciferase without any eukaryotic promoter or enhancer elements We then generated a series of 5¢-serial-deletion constructs of the promoter–luciferase gene for transfections and subsequent assays of luciferase activity (Fig 1A) Constructs D1 to D5, together with the pGL3-basic vector, were used for transient transfection of NIH3T3 cells in an attempt to identify the cis-elements of the gene for Dnmt1 and to delineate the 5¢ boundary of the promoter As shown in Fig 1A, weak control of promoter activity was associated with a region of

1827 bp in the upstream region between nucleotides)2000 and)173, with no more than 40% variation in activity A large reduction of approximately sixfold in promoter activity was detected when we removed nucleotides)173

to )120 (Fig 1A, D3 to D5), suggesting that cis-acting elements that are critical for Dnmt1 promoter activity might

be located in this region We detected a single site for initiation of the transcription of Dnmt1 at +1 in this promoter (data not shown), a result that is consistent with previous reports [20,23] Thus, it appeared that the region from nucleotides )173 to )120 contained critical cis-elements for transcriptional activation of the Dnmt1 gene

To examine DNA-binding proteins, we prepared DNA probes from the promoter of the mouse gene for Dnmt1 (nucleotides )173 to )117), which contained putative binding sites for Sp1, and performed gel shift assays as described previously [26] Three DNA probes, P1 (nucleo-tides)173 to )138), P3 (nucleotides )160 to )131)and P2 (nucleotides )141 to )117)were prepared (Fig 1B) EMSAs of nuclear extracts from NIH3T3 cells with the P1 probe revealed shifts of three bands on the gel (B1 to B3

in Fig 1C) The intensity of these bands was significantly enhanced with P3 probe and no DNA–protein complexes were evident when we analyzed nuclear extracts of NIH3T3 cells with the P2 probe We observed three similar shifted bands when we used nuclear extracts from 3T6 cells, HeLa cells, 293 cells and F9 cells (data not shown) To define the binding site in this region more precisely, we synthesized 11 mutant oligodeoxynucleotides (ODNs; M1 to M11 in

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Fig 2B)and used them as competitiors in EMSAs

(Fig 2A) The DNA–protein complexes B1, B2, and B3

were detected with the wild-type P1 probe After addition of

a 100-fold excess of unlabeled mutant ODN (M1 to M11)to

the reaction mixture, we found that shifted bands were not

eliminated by the unlabeled mutant ODNs M5 through M8

The nucleotide sequences of these ODNs were

com-pared and the consensus-binding site was defined as the

15-bp sequence 5¢-GGCAAGGGGGAGGTG-3¢ (Fig 2B),

which we designated the GA motif To examine whether or

not this sequence is critical for the transcriptional activity of

the Dnmt1 promoter, we synthesized 10 mutant ODNs

(n1 to n10 in Fig 2D)and introduced them into luciferase

constructs to generate the respective reporter plasmids We

examined the activity of these luciferase reporters in

NIH3T3 cells and found that only the n5 construct lacked

transcriptional activity (Fig 2C) Thus, the central GG

dinucleotide in the 15-bp motif seemed to be critical for the

transcriptional activity of the Dnmt1 promoter

Sp1 and Sp3 bind to the GA motif in the Dnmt1 promoter

To identify the transcription factors that bind to the GA

motif in the Dnmt1 promoter, we looked for transcription

factors in the TRANSFAC database [27] We failed to

identify known factors that bind to this sequence However,

we found that both AP-2 and Sp1 bound to sequences that

exhibited strong similarity, namely 0.859 and 0.856,

respectively, to the GA motif To determine whether Sp1

and/or AP-2 could bind to the GA consensus sequence, we

performed EMSAs in the presence of antibodies against

Sp1, Sp3 and AP-2 (Fig 3A) The retarded band designed

B1 was shifted even further upon addition of antibodies

against Sp1 (lane 3), while the retarded bands corresponding

to B2 and B3 were shifted further upon addition of

antibodies against Sp3 (lane 4) Both antibodies against Sp1

and against Sp3 affected the migration of all retarded bands

In contrast, anti-(AP-2)Ig did not affect the migration of

shifted bands (lane 6) Thus, it appeared that both Sp1

and Sp3 bound to the 5¢-GGCAAGGGGGAGGTG-3¢

sequence in the Dnmt1 promoter in vitro We also performed

a chromatin immunoprecipitation experiment to assess the

interaction of Sp1 and Sp3 in the transcription of the Dnmt1

gene at the chromatin level (Fig 3B) In contrast to the

absence of any effect of IgG, immunoprecipitates obtained

with antibodies both against Sp1 and against Sp3 yielded a

318-bp DNA fragment after PCR that included the 15-bp

sequence Antibodies against AP-2 did not generate the

318-bp DNA (data not shown) These results supported our

hypothesis that both Sp1 and Sp3 bind to cis-elements in the

gene for Dnmt1 that include the GA motif

Stimulation of transcription from the Dnmt1

promoter by Sp1 and Sp3 in SL2 cells

Both Sp1 and Sp3 bound to the Dnmt1 promoter

Therefore, we next examined the effects of Sp1 and Sp3

on the expression of the gene for Dnmt1 We transfected

DrosophilaSL2 cells with a luciferase reporter construct in

the presence and absence of the expression plasmids

pPacSp1 and pPacUSp3, respectively SL2 cells lack

endogenous Sp proteins, and, thus, the

cis-element-depend-ent activation of the Dnmt1 promoter is dependcis-element-depend-ent on the

gene products of exogenously introduced genes that encode Sp1 or Sp3 The activity associated with the luciferase reporter construct D2 was stimulated in the presence of pPacSp1 (Fig 4A)and in the presence of pPacUSp3

Fig 3 Sp1 and Sp3 bound to a cis-element in the promoter of the mouse gene for Dnmt1 (A)32P-Labeled probe P3 (1 · 10 4 c.p.m)was incu-bated with 5 lg protein of nuclear extract (NE)from NIH3T3 cells in the presence and absence of antibodies against Sp1, Sp3 or AP-2 Lane

3, Sp1-specific antibody (2 lg); lane 4, Sp3-specific antibody (2 lg); lane 5, Sp1-specific and Sp3-specific antibodies (2 lg each); lane 6, AP-2-specific antibody (2 lg) (B) Chromatin immunoprecipitation assays Chromatin immunoprecipitation assays were performed as described in the text DNA and proteins were cross-linked with formaldehyde, and DNA was sheared and immunoprecipitated with Sp1- or Sp3-specific antibody (2 lgÆmL)1each) After reversal of cross-links, DNA was amplified with primers specific for the promoter region of the Dnmt1 gene Products of PCR were resolved by agarose gel electrophoresis The arrowhead indicates the amplified DNA fragment (318 bp).

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(Fig 4B) Moreover, the promoter activity of D2 was

further enhanced in the presence of both pPacSp1 and

pPacUSp3 (Fig 4E) In contrast, the activity associated

with the luciferase reporter construct n5 was not stimulated

by either pPacSp1 or pPacUSp3 (Fig 4C,D) These results

indicate that both Sp1 and Sp3 enhanced transcription from

the Dnmt1 promoter

Independent activation of the Dnmt1 promoter

by Sp1 and Sp3 through the same GA motif

Sp1 and Sp3 bound to the same cis-element in the promoter

of the gene for Dnmt1 Therefore, we next examined

whether activation by Sp1 or by Sp3 affect expression of the

gene for Dnmt1 In an attempt to identify whether Sp1 and

Sp3 could bind to the same GA motif, we prepared

appropriate GST fusion proteins and performed EMSAs

with the P3 probe that included the GA motif Specific and

different shifted bands were detected with GST–Sp1 and

GST–Sp3 The shifted band due to GST–Sp3 gradually

disappeared when increasing amounts of GST–Sp1 were

added as competitor (Fig 5A,B) In contrast, the intensity

of the shifted band that included GST–Sp1 gradually

increased Conversely, the addition of GST–Sp3 resulted in

a similar reduction in the binding of Sp1 to the GA motif

(data not shown) These results imply that the binding of

Sp1 and of Sp3 to the GA motif were independent

phenomena and that each competed for binding to DNA with the other through the same cis-element

We next examined whether Sp1 might be included in transcriptional complexes with Sp3 in vivo Western blotting analysis with antibodies against Sp1 of immunoprecipitates obtained with antibodies against Sp3 indicated that Sp1 was not immunoprecipitated with Sp3 from extracts of NIH3T3 cells (Fig 5C) Complementary studies of immunoprecipi-tates obtained with Sp1-specific antibodies and Western blotting with antibodies against Sp3 also indicated that Sp1 and Sp3 did not interact with each other (data not shown)

We also examined the molecular association of GST–Sp1 and GST–Sp3 fusion proteins and found no evidence of any association in vitro (data not shown)

Enhancement by p300 of transcription from the Dnmt1 promoter that is induced by Sp3

The transcriptional coactivator p300 mediates growth arrest

by catalyzing histone acetylation and the subsequent rearrangement of chromatin [11,12] Recent reports indicate that p300 also collaborates with Sp1 or Sp3 to regulate the expression of the promoter of the gene for p21Waf1/Cip1 [28,29] Therefore, we examined the effect of p300 on the promoter activity of the Dnmt1 gene in the presence of pCMV-Sp1 and of pCMV-Sp3 in NIH3T3 cells As shown

in Fig 6, cotransfection with pCi-p300 and pCMV-Sp3

Fig 4 Activation of transcription from the Dnmt1 promoter by Sp1 and Sp3 in Drosophila SL2 cells SL2 cells were transfected with 500 ng of the Dnmt1 reporter plasmid D2 (A,B,E)and the reporter plamid n5 without GA motif (C,D)and the indicated amounts of pPacSp1 (A,C), pPacUSp3 (B,D)or both pPacSp1 and pPacUSp3 (E)(each 500 ng)and then luciferase activities were measured The total amount of the plasmid DNA (pPacSp1 or pPacUSp3)was adjusted to 1 lg with pPac (no insert) Assays were repeated at least three times and the standard deviation for each mean value is indicated.

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enhanced the reporter activity controlled by the Dnmt1

promoter, but cotransfection of pCi-p300 with pCMV-Sp1

did not The extent of activation was much higher than that

obtained with pCi-p300 and with pCMV-Sp3, indicating

that p300 enhanced the promoter activity of the Dnmt1 gene

that was induced by Sp3, but not by Sp1 Further studies of

transactivation using a GAL4 fusion with p300 and the

dominant negative form of p300 are required for a full

understanding of the molecular mechanism of this

phe-nomenon

Sp3 interacts with the C-terminal region of p300

To determine whether Sp3 associates directly with p300,

we performed immunoprecipitation and Western blotting

assays using antibodies against p300 and Sp1 or Sp3 and extracts of NIH3T3 cells We immunoblotted immunopre-cipitates obtained with p300-specific antibodies with anti-bodies against Sp1 or against Sp3 As shown in Fig 7A, a p300-specific band was detected with antibodies against Sp3 but not against Sp1 Antibodies specific for AP-2 (data not shown)and control IgG did not yield any evidence of interactions with Sp3 To determine which regions of p300 interacted with Sp3, we prepared 35S-labeled Sp3 by translation in vitro and investigated the binding to various GST–p300 fusion proteins by the GST-pull down assay [25] Only the C-terminal region of p300 (amino acids 1572–2414), which included the C/H region and E1A-binding region of p300, was found to associate with Sp3 (Fig 7B) We performed similar complementary experi-ments to determine which parts of p300 interacted with Sp3 and found that the [35S]Met-labeled carboxyl region of p300 interacted with GST–Sp3 (data not shown) These results indicated that Sp3 was able to bind to the C-terminal domain of p300

D I S C U S S I O N

The promoters of many housekeeping genes have a number of common characteristics, such as the presence

of multiple sites for initiation of transcription which, presumably, compensate for the absence of a TATA box and a CAAT box, and they often have an unusual high GC-content [1] The Dnmt1 gene is also a housekeeping

Fig 5 Binding of Sp1 and Sp3 to the cis-element in the promoter of the

Dnmt1 gene (A)EMSAs of GST-Sp1 and GST-Sp3 fusion proteins

with the P3 probe Indicated amounts of GST-Sp1 (onefold to 50-fold

excess)were added to a reaction mixture that included 0.1 lg of

GST-Sp3 and the P3 probe (0.2 nmolÆmL)1) (B) Relative binding of Sp1

and Sp3 to P3 in EMSA The results in (A)are summarized as relative

DNA-binding activity (%) The total intensity of P3 probe on a

reaction mixture for EMSAs was taken arbitrarily as 100% (C)Sp1

and Sp3 do not form a stable complex in NIH3T3 cells

Immuno-blotting of Sp1 in the immunoprecipitates derived from 500 lg protein

of whole-cell extracts of NIH3T3 cells with Sp3-specific antibody

(2 lgÆmL)1) Sp1 did not form a complex with Sp3 in NIH3T3 cells.

Input: this lane was loaded 50 lg protein of whole-cell extracts.

Fig 6 Enhancement of the Dnmt1 promoter activity upon cotransfec-tion with p300- and Sp3-expression plasmids NIH3T3 cells were transfected with 0.5 lg of the Dnmt1 gene reporter plasmid D2 (see Fig 1A)plus 0.5 lg of pCMV-Sp1, pCMV-Sp3 or/and pCi-p300 and luciferase activity was measured in each case Total amounts of DNA (pCMV-Sp1, pCMV-Sp3 and pCi-p300)were adjusted to 2 lg with pBSII KS(+) The assay was repeated at least three times and the standard deviation for each value is indicated.

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gene [24] However, details of the cis-elements in its

promoter and the factors that regulate expression of the

Dnmt1 gene from the minimum promoter in somatic cells

remain to be determined In this report, we identified a

cis-element in the promoter of the Dnmt1 gene in somatic

cells and showed that both Sp1 and Sp3 bound to this

regulatory element, the GA motif, independently

More-over, both Sp1 and Sp3 stimulated the promoter activity

of the Dnmt1 gene and Sp3 was found to associate with

p300 through the C-terminal region of the latter protein

and to enhance its activity

In order to identify the minimal promoter in a 2.0-kb

region of the Dnmt1 gene, we generated a series of

promoter–luciferase deletion constructs for reporter assays and then determined the luciferase activity associated with each respective construct (Fig 1A) The results indicated that a cis-acting region of 53-bp was located between nucleotides)173 and )120 Precise dissection of the region

by EMSAs demonstrated that a minimal element, from nucleotides)161 to )147, was critical for the activation of transcription of the Dnmt1 gene (Fig 2A) We identified a

GA motif in this region required for the binding of SP1 and SP3 to the DNA (Fig 3A,B)in order to activate transcrip-tion of the Dnmt1 gene (Fig 4) EMSAs in the presence of anti-Sp1 Ig and anti-Sp3 Ig confirmed these results (Fig 3A) The GA motif is different from the typical sequence of Sp1-binding sites, GGGCGG (Fig 2) Thus, Sp1- and Sp3-binding sites might be affected by sequences adjacent to GC-rich or GA-rich elements that influence maximum binding It has been reported that Sp1 and Sp3 might be involved in the activation of a very large number of genes, such as housekeeping genes for tissue-specific and cell cycle-regulated proteins [3] Moreover, Sp proteins are involved not only in activation but also in repression We studied the effects on the expression of the Dnmt1 gene by Sp1 or Sp3, which bound to the similar elements (Fig 3) The two proteins are found in the same cells and are indistinguishable in terms of DNA-binding specificity (Fig 5A,B), and both proteins bound independently to the GA motif (Fig 5C) All Sp proteins contain three zinc fingers close to the C-terminus, with glutamine-rich domains adjacent to serine/threonine structures in the N-terminal region Sp1, Sp3 and Sp4 are more closely related to each other than they are to Sp2 [3] The homology among the zinc fingers of all known Sp proteins is close to 90%, but, the homology among entire sequences is close to 40%

In the transcriptional activation of the Dnmt1 gene, both Sp1 and Sp3 play a critical role (Fig 4)via binding to the same cis-element (GA motif)and the binding of each is independent of the other (Fig 5) A competition experiment

in vitrowith the GA motif demonstrated that each factor is able to replace the other in terms of binding to DNA The growth characteristics of Sp1-deficient embryonic stem cells (ES cells)are normal and such cells can be induced

to differentiate [30] Nevertheless, Sp1 is essential for normal mouse embryogenesis and the development of Sp1-knock-out embryos is severely retarded, with death occurring around day 11 of gestation Thus, Sp1 appears to be a transcription factor whose function is essential after day 10

of development Other Sp proteins, such as Sp3, might be able to compensate, at least in part, for the loss of Sp1 activity at early embryonic stages Sp3 is expressed ubiqui-tously and has the potential to activate transcription Moreover, its DNA-binding activity is indistinguishable from that of Sp1 A recent study of Sp3 null-mice suggested that Sp1 and Sp3 might have similar and therefore redundant functions during early development but might have distinct and highly specific functions at later stages of development [31] Thus, Sp1 and Sp3 might have a wide range of redundant functions and might be able to replace each other in Sp1-and Sp3-knockout mice [31] It was reported that three isoforms of Sp3 exist and that these different isoforms play different roles in the activation and repression of transcription [9] In our hands, Sp1 generated

a single band and Sp3 generated two bands in EMSAs (two

of three bands migrated to the same position; B3 in

Fig 7 p300 was associated with Sp3 (A)p300 associated with Sp3 and

not with Sp1 in NIH3T3 cells p300 was immunoprecipitated with

anti-p300 Ig (2 lg)from 500 lg protein of whole-cell extract of NIH3T3

cells and the immunoprecipitate was immunoblotted with antibodies

against Sp3 (2 lgÆmL)1; upper panel)or Sp1 (2 lgÆmL)1; lower panel)

as described in the text (B)Direct interaction of Sp3 with various

GST–p300 deletion mutants 35 S-Labeled Sp3 (3 · 10 3 c.p.m)was

incubated with 2.0 lg of each GST–p300 mutant, as indicated (lanes

3–5), or with 2.0 lg GST alone (lane 2) Lane 1, one tenth of input

35 S-labeled Sp3 After electrophoresis, radiorabeled protein was

detected by autoradiography The shaded boxes of p300 indicate the

C/H1 domain, C/H2 domain and C/H3 domain [10].

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Fig 3A) It has been suggested that two small isoforms of

Sp3 might act as repressor molecules with full-length Sp3

acting as an activator [3,9] Sp3 acts as a transcriptional

activator at many promoters, as does Sp1 [5,32] In studies

of other promoters such as the uteroglobin gene [8],

monocyte chemoattractant protein-1 gene and ornithine

decarboxylase gene [7,33], however, Sp3 was found to be

inactive or to act as only a weak activator or as a repressor

of Sp1-mediated transcription

The most obvious differences between Sp1 and Sp3 are

the presence of a potent inhibitory domain in Sp3 [34] The

relative abundance of Sp1 and Sp3 allows the fine tuning of

the regulation of gene activities In endothelial cells that

contain high levels of both Sp1 and Sp3, the ratio of Sp1 to

Sp3 is higher than in nonendothelial cells [35] In primary

keratinocytes, levels of Sp3 exceed those of Sp1 The ratio of

Sp3 to Sp1 is inverted when these cells differentiate in vitro

In differentiating keratinocytes, only Sp3 enhances the

activation of the promoter of the gene for p21Waf1/Cip1[36]

A change in the ratio of Sp1 to Sp3 also occurs when C2C12

myocytes are cultivated under hypoxic conditions Hypoxia

causes the progressive depletion of Sp3, whereas the level of

Sp1 remains unchanged [37] It has been demonstrated that

the expression of the gene for Dnmt1 is regulated in a

cell-cycle-dependent manner The expression of Dnmt1 is

enhanced during the S phase and then declines with the

approach of the M phase In a preliminary study, we found

that levels of expression of Sp1 and Sp3 differed during the

cell cycle; Sp1 was expressed predominantly at the G1 phase

and Sp3 was expressed at S phase (data not shown)

Therefore, it is quite plausible that Sp1 and Sp3 might

control the expression of the Dnmt1 gene Further studies

are required to clarify the distinct roles of Sp1 and Sp3 at

different phases of the cell cycle

The transcriptional cofactor p300 is coprecipitated in

complexes with Sp1 [38] The activation of the promoter of

the gene for p21Waf1/Cip1 by butyrate and nerve growth

factor requires functional collaboration between Sp1 and

Sp3 [28,38] However, although p300 and Sp1 are

compo-nents of the complex that activates the promoter of the gene

for p21Waf1/Cip1, the interaction is indirect Thus, p300 is

required for the tricostatin A-induced (TSA-induced),

Sp1-mediated transcription of the gene, but details of the

interaction between Sp1 and p300 in this phenomenon are

unknown It is possible that Sp3 might bind to a GC or GA

motif in the promoter of the gene for p21Waf1/Cip1through

binding with p300, as such motifs might bind Sp1 and Sp3

Thus, Sp3 might regulate the TSA-dependent activity of the

promoter We showed that Sp3 might associate with the

C-terminal region of p300 by direct binding (Fig 7B) This

region is similar to the region to which GATA-1, E2F and

p53 also bind [10] Thus, regulation by Sp1 and Sp3 of target

genes might be involved in the cell cycle Furthermore, the

activity of p300 is dependent on gene dosage during early

embryogenesis Thus, it is possible that Sp3 might compete

for binding to DNA with Sp1-like proteins and

Kru¨ppel-like factors via interactions with p300 at different phases of

the cell cycle in somatic cells In conclusion, we propose that

the nucleotide sequences to which Sp1 and Sp3 bind are

similar and binding of these factors is independently

regulated by different coactivators in a cell-cycle-dependent

manner The distinct functions of Sp1 and Sp3 in the

regulation of expression of the Dnmt1 gene during the cell cycle remain to be clarified

A C K N O W L E D G E M E N T S The authors thank Drs G Suske, R Chiu, Y Shi, Y Nakatani,

S Kojima, H Ugai, C Jin, J Song and A Wolff for plasmids, reagents and valuable discussions This work was supported by the Special Coordination Funds of RIKEN, by grants from the Ministry of Education, Science, Sports, Culture and Technology of Japan (to

K K Y)and a grant from the Program for Promotion of Basic Research Activities for Innovative Biosciences (to K S).

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