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Tiêu đề Transactivation properties of c-Myb are critically dependent on two SUMO-1 acceptor sites that are conjugated in a PIASy enhanced manner
Tác giả ỉyvind Dahle, Tor ỉ. Andersen, Oddmund Nordgård, Vilborg Matre, Giannino Del Sal, Odd S. Gabrielsen
Trường học University of Oslo
Chuyên ngành Biochemistry
Thể loại báo cáo khoa học
Năm xuất bản 2003
Thành phố Oslo
Định dạng
Số trang 11
Dung lượng 266,96 KB

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Here we provide independent evidence that human c-Myb is also subject to SUMO-1 conjugation under more physiological condi-tions as revealed by coimmunoprecipitation analysis of Jurkat c

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Transactivation properties of c-Myb are critically dependent

on two SUMO-1 acceptor sites that are conjugated

in a PIASy enhanced manner

Øyvind Dahle1, Tor Ø Andersen1, Oddmund Nordga˚rd1, Vilborg Matre1, Giannino Del Sal2,3

and Odd S Gabrielsen1

1

Department of Biochemistry, University of Oslo, Norway;2Laboratorio Nazionale CIB, Area Science Park, Trieste, Italy;

3

Dipartimento di Biochimica, Biofisica e Chimica delle Macromolecole, Universita` degli Studi di Trieste, Italy

The transcription factor v-Myb is a potent inducer of

myeloid leukemias, and its cellular homologue c-Myb

plays a crucial role in the regulation of hematopoiesis

Recently, Bies and coworkers (Bies, J., Markus, J &

Wolff, L (2002) J Biol Chem, 277, 8999–9009) presented

evidence that murine c-Myb can be sumoylated under

overexpression conditions in COS7 cells when

cotrans-fected with FLAG-tagged SUMO-1 Here we provide

independent evidence that human c-Myb is also subject to

SUMO-1 conjugation under more physiological

condi-tions as revealed by coimmunoprecipitation analysis of

Jurkat cells and transfected CV-1 cells Analysis in an

in vitro conjugation system showed that modification of

the two sites K503 and K527 is interdependent A

two-hybrid screening revealed that the SUMO-1 conjugase

Ubc9 is one of a fewmajor Myb-interacting proteins The

moderate basal level of sumoylation was greatly enhanced

by cotransfection of PIASy, an E3 ligase for SUMO-1 The functional consequence of abolishing sumoylation was enhanced activation both of a transiently transfected reporter gene and of a resident Myb-target gene When single and double mutants were compared, we found a clear correlation between reduction in sumoylation and increase in transcriptional activation Enhancing sumoy-lation by contransfection of PIASy had a negative effect

on both Myb-induced and basal level reporter activation Furthermore, PIASy caused a shift in nuclear distribution

of c-Myb towards the insoluble matrix fraction We propose that the negative influence on transactivation properties by the negative regulatory domain region of c-Myb depends on the sumoylation sites located here Keywords: c-Myb; transcription; SUMO-1; Ubc9; PIASy

The c-Myb transcription factor plays a central role in the

regulation of cell growth and differentiation, in particular in

hematopoietic progenitor cells (reviewed in [1])

Homozy-gous null c-Myb/Rag1 chimerical mice are blocked in early

T-cell development, while mice with a c-mybnull mutation

display severe hematopoietic defects leading to in utero

death at E15 [2,3] The c-Myb protein consists of an

N-terminal DNA-binding domain (DBD), a central

trans-activation domain (TAD) and a C-terminal negative

regulatory domain (NRD) The DBD of c-Myb is

com-prised of the three imperfect repeats: R1, R2and R3, each

related to the helix-turn-helix motif [4–7]

Oncogenic alterations, as found in AMV v-Myb, include

both N- and C-terminal deletions as well as point mutations

[8] AMV v-myb is a potent and cell-type specific oncogene that transforms target cells in the macrophage lineage and induces monocytic leukemia [8,9] Several studies have attempted to define oncogenic determinants of v-myb N- and C-terminal deletions remove several sites of protein modification, including an N-terminal CK2 phosphoryla-tion site (S11 and S12) [10], and a putative MAPK-site (S528) [11–13] as well as acetylation sites [14,15] located in the deleted portion of the C-terminal NRD In addition, specific point mutations in v-Myb abolish protein–protein interactions [5], as well as phosphorylation as in the case of V117D [16] c-Myb has recently also been reported to be subjected to SUMO-1 (small ubiquitin-related modifier) conjugation [17]

The SUMO-1 protein is related to ubiquitin, but its function, although presently unclear, seem to be other than proteasomal degradation (reviewed in [18,19]) The sequence homology between ubiquitin and SUMO-1 is low, but the structures are highly similar [20], and they use related conjugation mechanisms [21], including the use of E3-like factors, which was recently identified for sumoylation as the PIAS proteins (protein inhibitor of activated STATs) [22–25] The sequence YKXE has been proposed as a consensus sequence for SUMO-1 conjugation [26] The process of sumoylation is conserved from yeast to man and is a dynamic and reversible

Correspondence to O S Gabrielsen, Department of Biochemistry,

University of Oslo, PO Box 1041 Blindern, N-0316 Oslo, Norway.

Fax: + 47 22 85 44 43, Tel.: + 47 22 85 73 46,

E-mail: o.s.gabrielsen@biokjemi.uio.no

Abbreviations: DBD, DNA-binding domain; MRE, Myb recognition

element; NRD, negative regulatory domain; PIAS, protein inhibitior

of activated STATs; SUMO-1, small ubiquitin-related modifier;

TAD, transactivation domain; Ubc9, ubiquitin conjugation enzyme 9.

(Received 5 December 2002, revised 31 January 2003,

accepted 6 February 2003)

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process Both sumoylation and desumoylation are needed

for viability in yeast [27]

Because several different classes of proteins are targets for

SUMO-1 conjugation, it is rather unlikely that a single

explanation for the biological role of sumoylation will be

found A more general proposition is that sumoylation plays

a role in the stabilization of higher order protein complexes

and modification of protein–protein interactions [18] This is

consistent with the role of sumoylation in PML nuclear

bodies where it is important for PML nuclear body dynamics

and for recruiting other nuclear body components [28]

In the present study we have extended the findings of Bies

et al [17] by providing several lines of independent evidence

for this novel modification of c-Myb We showthat c-Myb

interacts strongly with Ubc9 causing sumoylation at two

specific sites in the NRD region of the protein, K527 being a

dominant site and K503 being secondary When

sumoyla-tion was blocked by mutasumoyla-tion of the two modificasumoyla-tion sites,

this caused a large increase in transcriptional activity of

c-Myb, both when assayed with a transiently transfected

reporter gene and when measuring a resident Myb-target

gene SUMO-1 conjugation was significantly enhanced by

cotransfection with PIASy, which is the E3 like factor

reported to enhance sumoylation of LEF1 [29]

Further-more, PIASy seems to increase the fraction of Myb species

in the insoluble part after subnuclear fractionation, which

indicates that sumoylation might be involved in modulating

the protein–protein interactions of c-Myb

Materials and methods

Plasmids

The yeast bait plasmid pDBT-hcM encoding full-length

human c-Myb fused to the Gal4p DBD was generated

from a cDNA clone [30] and the vector pDBT [31] The

mammalian expression plasmid pCIneo-hcM contains

full-length human c-Myb cDNA with an optimized ATG

context A c-Myb-HA fusion cDNA was generated by

cloning oligos encoding the C-terminal part of c-Myb in

fusion with an HA-tag, between PshAI and SalI in

pCIneo-hcM, to give the plasmid pCIneo-hcM-HA The

cDNAs encoding c-Myb mutants K503R, K527R and

K503/527R (abbreviated 2KR) were generated using the

Quick Change Site-Directed Mutagenesis Kit (Stratagene)

on a subfragment of human c-MYB Plasmids expressing

full-length SUMO-1 with an HA epitope (HA-SUMO-1)

or in fusion with GFP (GFP-SUMO-1) have been

described [32] The expression plasmid pGEX-UBC9 was

constructed from a human UBC9 cDNA (isolated in the

two-hybrid screening), and cloned in-frame into

pGEX-6P-2 between SalI and NotI All cloned fragments

generated by PCR were verified by sequencing The

c-Myb-responsive luciferase reporter construct pGL2/tk/

3xGG contains multimerized Myb response elements and

its construction is described in [33]

Yeast two-hybrid screen

The yeast two-hybrid screen was performed in the yeast

strain PJ69-4a [34,35] with pDBT-hcM as bait and using

two Matchmaker cDNA libraries (Clontech): from human

bone marrow(HL4053AH) and from the erythroleukemia cell line K562 (HL4032AH)

Cell culture, transfections and luciferase assays CV-1 and HD11 cells were grown as described [33,36] Transient transfections were performed by lipofection (Lipofectamine-Plus, Gibco Life Technologies) or using Fugene (Roche Diagnostics) Luciferase assays were per-formed in triplicate using the Luciferase Assay Reagent (Promega) Data from three independent transfection experiments were normalized for protein concentration in the samples Equal transfection efficiency was verified by Western analysis of the transfected species

In vitro conjugation assay The various forms of human c-Myb were generated in the TNT rabbit reticulocyte lysate system (Promega) in the presence of [35S]methionine Templates used were either the appropriate plasmid (pCIneo-hcM) or a PCR product with T7 promoter added during amplification (ÔTpC-fragmentÕ: amino acids 410–639) GST-SUMO-1 [37] and GST-UBC9 were expressed and affinity-purified using standard methods (Amersham Pharmacia Biotech) SUMO-activating enzyme (E1 fraction) was prepared from CV1 cells as described [37] SUMO-1 conjugation assays were performed as described

in [32] with purified GST-UBC9 included and incubation for two hours at 30C Reaction mixtures were analysed on 10% polyacrylamide gels revealed by fluorography Antibodies

For Myb detection, we used the polyclonal antibody H141 (Santa Cruz) and the monoclonal antibody 5e11 [38] SUMO-1 was detected with monoclonal antibodies from Zymed PIASy-T7 was detected using anti-T7 Ig (Novagen) Immunoprecipitation and Western blot

CV-1 cells were transfected with the indicated plasmids to analyse sumoylation of c-Myb After transfection, cells were lysed and subjected to coimmunoprecipitation as described [32] using standard methods

RNA isolation and real time PCR Total RNA was extracted from transfected HD11 cells using Absolutely RNATMRT-PCR Miniprep kit (Strata-gene) RNA (1–2 lg) was reverse transcribed with Super-script II reverse tranSuper-scriptase (Life Technologies) The cDNA was diluted fivefold prior to PCR amplification using primers specific for chicken mim-1 and chicken GAPDH, respectively Real-time PCR was performed on

a LightCycler rapid thermal cycler system (Roche Diag-nostics) using the LightCycler FastStart DNA Master SYBR Green I mix for amplification (Roche Diagnostics) Reactions were performed in 20 lL with 0.5 lM primers and 3 mMMgCl2 The amplification specificity of the PCR products was confirmed by using melting curve analysis and gel electrophoresis We calculated the relative level of mim-1 mRNA as 100/E(CP1–CP2), where CP1 and CP2 are crossing

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points for mim-1 and GAPDH mRNAs, respectively, and E

is the average efficiency of amplification obtained with the

same primer sets on a positive control template The mRNA

levels of GAPDH were thus set at 100%

Nuclear matrix preparation

CV-1 cells seeded out in 10 cm Petri dishes were transfected

with the indicated plasmids Cells were harvested 24 h after

transfection in NaCl/Pi and 30% were lysed directly in

loading buffer as a control for transfection Nuclear matrix

samples and soluble fractions were prepared essentially as

described in [39]

Results

Bies et al [17] have shown that murine c-Myb can be

sumoylated under overexpression conditions in COS7 cells

when cotransfected with FLAG-tagged SUMO-1 The

conjugation sites were mapped to the NRD region of the

protein This work raised several questions that we have

addressed in a parallel study focusing on human c-Myb

Several lines of independent evidence for sumoylation

of c-Myb

Our first interest was to find independent evidence for this

novel type of post-translational modification of c-Myb to

better establish its physiological relevance In particular, we

were concerned by the overexpression conditions exclusively

used in the previous work on sumoylation of c-Myb [17]

We therefore initially performed a cotransfection

experi-ment similar to those reported by Bies et al [17] but

replacing the COS cells with CV-1 cells, known to cause less

amplification of transfected plasmids than COS cells [40]

When CV-1 cells were cotransfected with constructs

expressing human c-Myb and GFP-tagged SUMO-1, two

retarded doublet bands were observed (Fig 1A, lane 3)

These totally disappeared when the two putative

sumoyla-tion sites (in human c-Myb K503 and K527) were both

mutated (Ô2KR-MybÕ, Fig 1A, lane 9) Single mutations

K503R and K527R had intermediary effects, with a strong

reduction with K527R and less effect with K503R where

only the upper doublet disappeared (Fig 1A, lanes 7 and 5)

The same doublets of bands were seen in the control lanes 2

and 4 due to endogenous SUMO-1 Sumoylation at two

sites in c-Myb would be expected to generate two retarded

simple bands Hence, the doublets probably represent

c-Myb with one and two conjugated SUMO-1 moieties,

respectively, combined with or without a second type of

modification (such as phosphorylation) affecting migration

This confirms the observations of Bies et al [17] under more

moderate conditions of overexpression

To increase the stringency further we performed a similar

experiment in the absence of transfected SUMO-1 to see

whether endogenous levels of the peptide and its

conjuga-tion enzymes were sufficient to cause sumoylaconjuga-tion This

experiment was similar to what is shown in the control lanes

2, 4 and 6 in Fig 1A but the use of a higher exposure allows

the effects of the mutants to be more evident Again shifted

Myb-bands were observed in addition to the main 75 kDa

band (Fig 1B, lane 2), although the mobility shifts now

were more modest, consistent with conjugation of untagged SUMO-1 The K503R mutant caused the upper doublet to disappear (Fig 1B, lane 3) The K527R mutant caused a much more important reduction in intensity of the slower migrating forms (Fig 1B, lane 4) In this mutant, only a single additional band is seen, probably due to a less efficient sumoylation of the remaining K503 site Again, the 2KR mutant showed no retarded bands (Fig 1B, lane 5) To

Fig 1 Human c-Myb is sumoylated in residues 503 and 527 (A) CV-1 cells transfected with the Myb-expressing plasmids as indicated, and in addition with (+) or without (–) pGFP-SUMO-1 The Myb proteins expressed were full-length human c-Myb (hcM) and c-Myb mutated in lysine 503 (K503R) or 527 (K527R) or both (2KR) Cells were lysed directly in loading buffer before separation on SDS/PAGE and immunoblotting revealed by a monoclonal anti-Myb Ig (5E11) (B) CV-1 cells transfected with empty pCIneo vector (v) or plasmids expressing indicated Myb proteins as in (A) Cell lysates were subjected

to direct immunoblot with monoclonal anti-(c-Myb) Ig (C) CV-1 cells were transfected as in (B) Immunoprecipitation was performed with monoclonal SUMO-1 Ig (upper panel) and polyclonal anti-(c-Myb) Ig (lower panel) After SDS/PAGE the blot was revealed by mAb 5E11 (D) Cell lysates from Jurkat cells expressing endogenous c-Myb, was subjected to immunoprecipitation with polyclonal anti-HA Ig, polyclonal anti-(c-Myb) Ig and polyclonal anti-(SUMO-1)

Ig After SDS/PAGE of the immunoprecipitates, immunoblot analysis was performed using monoclonal anti-(c-Myb) Ig The arrow indicates the migration of unmodified c-Myb.

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verify that the observed modifications were indeed due to

SUMO-1 conjugation we performed at

coimmunoprecipi-tation experiment While the lysate from CV-1 cells

trans-fected with wild type c-Myb contained modified Myb-forms

that became immunoprecipitated with the anti-SUMO-1 Ig

(Fig 1C, lane 2), this was not the case with the 2KR mutant

(Fig 1C, lane 3) This supports that wild type c-Myb

becomes conjugated with SUMO-1, and that this

modifi-cation is abolished in the 2KR mutant Both variants of

c-Myb were equally expressed (Fig 1C)

Having shown that c-Myb is sumoylated in CV-1 cells by

endogenous levels of the conjugation machinery, we finally

addressed whether the same was true for endogenous c-Myb

proteins in myb-positive cells Therefore we carried out a

similar analysis in Jurkat cells Immunoprecipitation of

c-Myb with polyclonal anti-(c-Myb) Ig, and detection with

monoclonal anti(-c-Myb) Ig revealed the main c-Myb band

at 75 kDa and several c-Myb species with higher molecular

mass (Fig 1D, lane 2) Immunoprecipitation of sumoylated

proteins with polyclonal anti-(SUMO-1) Ig in the same

experiment revealed that at least one of these bands are

sumoylated c-Myb This was also confirmed by

immuno-precipitation of c-Myb with polyclonal anti-(c-Myb) Ig and

detection with monoclonal anti-(SUMO-1) Ig, which

revealed one band with a size corresponding to c-Myb

conjugated with one SUMO-1 molecule (results not shown)

We conclude that the SUMO-1 conjugation c-Myb

observed by Bies et al [17] under overexpression conditions

seems to be a robust phenomenon that also occurs under

more physiological conditions

A second line of experiments further supported that

c-Myb is a good substrate for SUMO-1 conjugation An

in vitrosystem for sumoylation was set up to investigate

sumoylation of c-Myb (Fig 2) When in vitro translated

human c-Myb was incubated with an E1 fraction,

GST-UBC9 and GST-SUMO-1, two more slowly migrating

forms were generated with sizes corresponding to the

addition of one or two moieties of GST-SUMO-1,

respect-ively (+39 kDa and +78 kDa) (Fig 2, lane 4) These

modified forms disappeared when either GST-UBC9 or

GST-SUMO-1 was omitted from the reaction mixture

(Fig 2, lanes 3 and 5), strongly suggesting that they

correspond to c-Myb conjugated to SUMO-1 peptides

Both retarded bands observed with the wild type protein

disappeared when the double mutant (2KR) was subjected

to in vitro sumoylation, demonstrating their function as

conjugation sites (Fig 2, lanes 7 and 9) Consistent with the

location of K503 and K527 in a region that is deleted in

AMV v-Myb, an AMV v-Myb protein did not generate

retarded modified forms in this system (results not shown)

The two single mutants, K503R and K527R, and the 2KR

mutant were also subjected to in vitro sumoylation in the

context of a c-Myb fragment (amino acids 410–566, more

efficiently translated in vitro) When the conjugated forms of

the single mutants were compared, it was evident that the

two sites were not equivalent While the K527R mutation

caused a sharp drop in sumoylation efficiency, requiring a

high input of UBC9 to become sumoylated on the

remaining site, the K503R protein was still efficiently

sumoylated at lowinputs of UBC9 similar to wild type This

strongly suggests that K527 is a much more efficiently

conjugated site than K503 It is also noteworthy that

bis-sumoylated wild type protein (modified in K503 and K527) is formed as efficiently as mono-sumoylated (pre-sumably mainly modified in K527), while mono-sumoylated K527R protein (presumably modified in K503) is formed with low efficiency This suggests that K527-conjugation enhances the efficiency of sumoylation at the other site

A third line of independent evidence for sumoylation of c–Myb is the interaction between c-Myb and Ubc9, the latter acting as an E2-type SUMO-1 conjugase Assuming such an interaction, Bies et al [17] performed a direct two-hybrid test for this interaction between Ubc9 fused to Gal4p-DBD and c-Myb domains fused to Gal4p-TAD Both fusion proteins were expressed from high-copy yeast vectors In an independent series of experiments we set up a two-hybrid screen using full-length human c-Myb as bait fused to Gal4p-DBD, but in our case expressed from a low-copy CEN vector Screening of 4· 106transformants from two mixed cDNA Matchmaker libraries (human bone marrowand human erythroleukemia K562 cell line) resulted in the isolation of 23 triple-positive independent clones Three of these were identical to mRNA for human ubiquitin-conjugating enzyme UBC9 (Accession

no AJ002385) Retransformation and growth on reporter-selective media (not shown) verified the Myb–UBC9 interaction, and by determination of reporter activation using both a 5-bromo-4-chlorindol-3-yl b-D-galactoside overlay and a liquid b-galactosidase assay (Fig 3) Similar analysis of several subdomains of c-Myb revealed strongest subdomain interaction with the EVES-domain in the NRD-region of c-Myb, suggesting that this NRD-region might be involved in the UBC9 interaction (results not shown) These two-hybrid results show that Ubc9 is amongst the strongest interaction partners of c-Myb as judged by a low-copy bait screening in a cDNA library containing 2 million inde-pendent clones, lending further support to the importance

of the c-Myb–Ubc9 interaction

We conclude that SUMO-1 conjugation of c-Myb is not only a phenomenon induced under favourable conditions of overexpression of c-Myb and SUMO-1, but a robust modification caused by a strong interaction between c-Myb and Ubc9 This leads to modification at two residues

in the NRD part of the protein with K527 being the major sumoylation site The conjugation of SUMO-1 to c-Myb raises the question of the role of this modification with respect to the transcriptional activity of c-Myb

Disruption of the SUMO-1 acceptor sites in c-Myb causes a superactivation phenotype

Bies et al [17] observed that c-Myb mutated in one of the sumoylation sites was more active than wild type Myb in an effector-reporter assay under overexpression conditions in COS7 cells To confirm this observation in CV-1 cells and

to extend the analysis to clarify the relative functional importance of the two conjugation sites, we compared reporter activation induced by the individual mutants (K503 and K527), the double mutant (2KR) and wild type c-Myb using a reporter with multimerized Myb response elements (Fig 4A) While full-length c-Myb caused a modest level of reporter activation (1.3-fold relative to empty effector), the K503R mutant was slightly more active (3.6-fold), the K527R mutant significantly more active (9.6-fold) and

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finally the double mutant c-Myb-2KR gave rise to a 23-fold

increase in reporter activity, which is 17-fold higher than

wild type c-Myb A Western blot confirmed that all Myb

variants were equally expressed (Fig 4A) It is noteworthy

that a clear correlation seems to exist between the increase in

transcriptional activity of the individual mutants (Fig 4A)

and the reduction in their degree of sumoylation (Fig

1A,B)

Because effector-reporter assays in transfected cells is a method with recognized limitations, we wanted to see whether the conjugation sites influenced transcriptional activity in a more physiological setting and therefore tested activation of the resident mim-1 gene using the HD11 cell line, an established Myb-model [36] The mim-1 target gene

is only activated by c-Myb, not by v-Myb, when residing in its chromosomal locus because v-Myb has lost the ability to

Fig 2 Human c-Myb is sumoylated by UBC9 in vitro (A) In vitro translated35S-labelled full-length c-Myb was incubated in the presence (+) or in the absence (–) of the indicated components described in Materials and methods (lanes 1–5) Single sumoylated (1· Sumo) and bis-sumoylated (2· Sumo) Myb, respectively In lanes 6–9 full-length c-Myb (hcM) and c-Myb mutated at K503 and K527 (2KR) were compared in the presence (+)

or absence (–) of the full set of sumoylation components (B) Wild type or mutant (K503R and K527R) subdomains of human c-Myb (TpC fragments, amino acids 410–639) were 35 S-labelled in vitro and subjected to sumoylation as in 2A, but with variable limiting amounts of GST-UBC9

as indicated (given as ng of UBC9 only) The amount of sumoylated Myb species were quantified by the NIH IMAGE 1.62 software (upper panel) and the different sumoylated Myb species measured are shown in the lower panel ÔWt bis-SÕ and Ôwt mono-SÕ represent double and single sumoylated wild type TpC c-Myb, respectively; ÔK503R mono-SÕ, single sumoylated K503R TpC c-Myb; ÔK527R mono-SÕ, single sumoylated K527R TpC c-Myb.

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cooperate with C/EBPb/NF-M, which is constitutively

expressed in these cells [5,41] When assayed by real-time

PCR, the AMV version caused only a marginal mim-1

activation, while c-Myb induced a significant level of mim-1

expression (Fig 4B, lanes 1 and 7) In this assay the 2KR

double mutant (lane 3) induced mim-1 expression to a

fourfold higher level than did wild type c-Myb (lane 1)

Cotransfection of SUMO-1 did not significantly change this

difference in behaviour, probably as the endogenous level of

SUMO-1 was already high and did not increase much after

SUMO-1 transfection (data not shown) Taken together,

these data clearly demonstrate that the conjugation sites in

K503 and K527 are critical for the potency of c-Myb to

activate the expression of a resident chromosomal c-Myb

target gene

PIASy enhances sumoylation of c-Myb and its

association with the nuclear matrix

Conjugation of SUMO-1 to target proteins has recently

been found to involve E3 enzymes in the PIAS family [22–

25] PIASy has been reported to enhance conjugation of

SUMO-1 to LEF1 [29] Based on this observation, we tested

whether PIASy also enhanced sumoylation of c-Myb, as

both LEF1 and c-Myb have been reported to be important

for differentiation in the hematopoietic system [42] As

shown in Fig 5, increasing amounts of transfected PIASy

caused a parallel increase in the intensity of the retarded

c-Myb species corresponding to single and double

sumoyl-ated c-Myb (Fig 5, lanes 1–4) No corresponding enhanced bands were observed with the 2KR mutant (Fig 5, lane 5) Thus, PIASy enhances conjugation of SUMO-1 to c-Myb, and probably functions as an E3 enzyme for this process Nowbeing able to greatly enhance the fraction of conjugated Myb molecules, we asked how this affected Myb-dependent transactivation, using a luciferase reporter

in transfected CV-1 cells As expected, the input of PIASy down-regulated Myb-dependent reporter activation was increased, but it turned out that PIASy expression caused a

Fig 4 Mutation of SUMO-1 conjugation sites enhances c-Myb acti-vity (A) Luciferase assays were performed on lysates from CV-1 cells transfected with plasmids encoding the indicated proteins, abbreviated

as in the legend to Fig 1, and a Myb-responsive reporter plasmid An aliquot of the lysates was analysed by Western blotting with anti-Myb

Ig to confirm expression of transfected Myb (lower panel) Note that standard lysis was used without precaution to avoid desumoylation upon cell lysis, hence less shifts are seen than in Fig 1B (B) The indicated plasmids were transfected into HD11 cells and total RNA was isolated Activation of the endogenous Myb-target gene, mim-1, was measured by real time PCR as described in Materials and meth-ods Abbreviations are as above and also ÔAMV vMÕ, AMV v-Myb; ÔSÕ, cotransfected with a SUMO-1 expressing plasmid pCDNA3-HA-SUMO-1.

Fig 3 UBC9 is a major Myb-interacting protein UBC9 was found

three times among 23 triple-positive independent clones isolated in a

yeast two-hybrid screening with full-length human c-Myb as bait The

UBC9/c–Myb interaction was verified as shown Right panel: Empty

library vector (pACT2) and pACT2-UBC9 were transformed into the

yeast two-hybrid strain PJ69-4a Similarly, empty bait vector (pDBT),

bait plasmid expressing lamin (pLam) and full-length human c-Myb

(pDBT-hcM-FL) were transformed into the a-mating type of the same

strain Mating was performed to create the diploid combinations

indicated in the figure These were subjected to

5-bromo-4-chlorindol-3-yl b- D -galactoside overlay assay to reveal activation of the LacZ

reporter gene as blue colour Left panel: PJ69–4a cells transformed

with plasmids encoding UBC9 fused to GAL4-AD (pACT2-UBC9),

c-Myb fused to GAL4-DBD (pDBT-hcM) and the two corresponding

empty vectors in the indicated combinations LacZ reporter activity

was measured by a liquid b-galactosidase assay The results are shown

as mean values ± SEM of four independent experiments, each carried

out in triplicate.

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parallel decrease in the basal activity of the reporter in the

Myb-negative controls (data not shown) This general

negative effect precluded any definitive conclusions from

this experiment as to whether SUMO-1 conjugated Myb is

transcriptionally less active than nonconjugated

To investigate additional consequences of

PIASy-enhanced sumoylation of c-Myb, we examined whether

the partitioning of c-Myb within the nucleus was altered

This hypothesis was based on the fact that sumoylation

modulates the protein–protein interactions between PML

and its protein partners [28,43] and that PIASy is localized

to the nuclear matrix [29] Therefore, we examined whether

PIASy-enhanced sumoylation of c-Myb had a general effect

on the interactions of c–Myb with other proteins in the

nucleus by doing a nuclear matrix (M) preparation

experi-ment as described in [39] This experiexperi-ment was done by

transfecting CV-1 cells with either c-Myb alone or c-Myb

together with PIASy When c-Myb was expressed alone, a

large portion of the c-Myb species was in the soluble

fraction (S) compared to the M-fraction (Fig 6, lanes 5 and

4) Sumoylated c-Myb was only visible here when a

comparable sample of the cells was lysed directly in the

loading buffer (lane 6) In contrast, coexpression with

PIASy resulted in a distinct change in the distribution of

c-Myb, with more c-Myb retained in M than found in S

(lanes 1 and 2) The sumoylated c-Myb species appeared to

accumulate preferentially in the M-fraction However,

because unsumoylated c-Myb species were also detected in

M (Fig 6, lanes 1 and 4), as was a significant fraction of the

2KR mutant (not shown), sumoylation cannot solely be

responsible for recruiting c-Myb to the nuclear matrix

fraction It is equally likely that PIASy somehowcauses

accumulation of c-Myb in the nuclear matrix and enhances

its sumoylation there, which then stabilizes its association

with the M-fraction We also noticed that the amount of

sumoylated c-Myb was not maintained during the

prepar-ation of M, which probably is due to sumoylprepar-ation being a

reversible process (Fig 6, T, M and S) This might give a larger fraction of unsumoylated c-Myb in M than is actually the case in vivo

We conclude that PIASy enhances sumoylation of c-Myb significantly, and that sumoylated and unsumoylated c-Myb showdifferences in intranuclear distribution, prob-ably as a consequence of altered protein–protein inter-actions between c-Myb and its protein partners Such differences might also be implicated in the increased activity

of 2KR compared to wild type c-Myb, but the mechanisms for this remain unidentified

Discussion

In the present study we have shown that the human transcription factor c-Myb is subject to conjugation by the small ubiquitin-related modifier, SUMO-1, at two sites in the NRD region of the protein, K527 being a principal sumoylation site and K503 a secondary one Both sites are important for transcriptional activity and their mutation causes a large enhancement of Myb-dependent transactiva-tion Sumoylation of c-Myb was strongly enhanced by coexpression of PIASy, which is the E3-like factor reported

to enhance sumoylation of LEF1 [29] This E3-induced increase in sumoylation also caused a shift in the distribu-tion of Myb species towards the insoluble fracdistribu-tion after subnuclear fractionation

Bies et al [17] recently reported that sumoylation of murine c-Myb can be induced by overexpression in COS7 cells of both c-Myb and FLAG-tagged SUMO-1 Here, we have reported a related study on human c-Myb that not only confirms the findings of Bies and coworkers, but also addresses several questions not answered by the previous study In particular, we were concerned that the modifica-tion had only been strictly demonstrated by cotransfecmodifica-tions

in COS cells This cell line is well known to cause amplification of effector plasmids containing an SV40

Fig 6 PIASy recruits wild type c-Myb to the nuclear matrix CV-1 cells were transfected with plasmids expressing wild type c-Myb alone

or in combination with PIASy as indicated The same number of cells from both transfections was used for nuclear matrix preparation as described in Materials and methods The cell suspension was divided

w ith one third used for preparation of the total fraction (T) and the remaining two-thirds used to make the soluble and nuclear matrix fraction Total protein concentration of the soluble fraction was used

to normalize between the preparations After separation of the proteins

on 10% SDS/PAGE, c-Myb species in the soluble (S), insoluble (M), and total (T) fractions were detected with immunoblotting using anti-(c-Myb) Ig as in Fig 1.

Fig 5 PIASy enhances sumoylation of c-Myb A plasmid expressing

wild type c-Myb was cotransfected into CV-1 cells with increasing

amounts of plasmid expressing PIASy (0 lg, 0.25 lg, 0.5 lg and

1.0 lg) as indicated As negative control 2KR c-Myb was

cotrans-fected with 1.0 lg PIASy plasmid The upper panel shows immunoblot

(IB) detection with anti-(c-Myb) Ig, and the lower panel shows PIASy

expression revealed with anti-T7 Ig and IB detection.

Trang 8

origin of replication (here pcDNA3-derived) due to the

presence of SV40 large T-antigen [40], which will lead to

significantly increased levels of expression In the present

study, we have tried to overcome these limitations and

provide three lines of independent evidence that human

c-Myb is indeed subject to SUMO-1 conjugation: (a)

immu-noprecipitations and Western analysis of Jurkat cells and

transfected CV-1 cells confirms that the modification

occurs at K503 and K527 under more physiological

conditions than previously reported, (b) analysis of

sumoy-lation in an in vitro conjugation system shows that c-Myb is

a good substrate for SUMO-1 conjugation and that

modification of the two sites are interdependent, and (c) a

two-hybrid screening shows that the SUMO-1 conjugase

Ubc9 is one of a fewmajor Myb-interacting proteins

expressed in bone marrowor erythroleukemia cell lines

We believe these independent data are important to be

confident that this novel type of modification of c-Myb is a

relevant one

The two sites in c-Myb became conjugated with unequal

efficiency, K527 being a principal sumoylation site and

K503 a secondary one, despite both having identical core

sequence motifs IKQE It is possible that the presence of

prolines close to K527 creates a more favourable context at

this site [44] The difference is clearly seen by the dissimilar

effects of mutations in the two sites The K527R mutant was

severely reduced in sumoylation in vivo (Fig 1A,B) and a

poor substrate in vitro compared to the K503R mutant

(Fig 2C), despite both harbouring one remaining

conjuga-tion site The large difference in efficiency could mean that

the K527 site is the only physiologically relevant site, as

indicated by the observation that endogenously expressed

c-Myb in Jurkat cells was detected with only one SUMO-1

peptide conjugated (Fig 1D) We cannot exclude, however,

that the two sites have distinct properties and that the

sumoylation of them depends on the biological context or is

controlled by specific E3 enzymes It has recently been

reported that PML harbours two independent sumoylation

sites with distinct properties [45] It is also possible that a

stepwise addition occurs The UBC9-titration experiments

in vitro suggested that K503-sumoylation occurred more

efficiently if K527 was already modified As SUMO-1 seems

to bind E3-type proteins [22], a possible scenario is that the

strong K527 is modified first, followed by enhanced

recruitment of an E3 activity through binding to SUMO-1

causing more efficient modification of the remaining weaker

site (although E3 was not added in vitro, a rather crude

source of E1 was used)

An important novel finding is that the modest level of

modification observed in continuously growing cells is not

constitutive but can be enhanced significantly upon a

change in the level of a specific E3 enzyme (Fig 5) PIASy,

the E3-like factor reported to enhance sumoylation of LEF1

[29], was found to drastically enhance sumoylation of

c-Myb on both sites This E3-induced increase also caused a

shift in distribution of Myb species towards the insoluble

fraction after subnuclear fractionation, as discussed below

The functional implications of c-Myb being prone to

SUMO-1 conjugation are not yet fully understood One

obvious possibility is regulation of transcriptional

activa-tion Sumoylation appears to have an effect on the activity

of several transcription factors, such as p53 [26,32], c-Jun

[46], Lef1 [29], AR [47], Sp3 [48], IRF-1 [49] and HDAC4 [50] Sp3 is a particularly illustrative example of a factor where SUMO modification, as with c-Myb, silences tran-scriptional activity Analysis of several mutations in Sp3 showed that those that prevented SUMO modification all strongly enhanced the transcriptional activity of the factor [48] Similarly, SUMO-1 conjugation in c-Myb occurs at sites that are very important for the activity of the factor Even a conservative mutation (KfiR) keeping the charge unchanged, causes a large enhancement of the activity of c-Myb both in transfection assays and w hen activation of an endogenous target gene is monitored That the relative enhancements were different in the two systems tested certainly relates to the many differences between the two cellular assays, including the use of a synthetic promoter (multimerized Myb response elements) vs a chromatin embedded target gene, different cooperation between fac-tors on the two promoters, and cofacfac-tors present in the hematopoietic cell line not present in CV-1 cell line When single and double mutants were compared we observed a clear correlation between the increase in transcriptional activity of the individual mutants (Fig 4) and the reduction in their degree of sumoylation (Fig 1A,B) Most probably these differences are caused by abolished sumoylation, which alters the transactivation properties Such changes could occur directly, by modula-tion of Myb’s intrinsic activamodula-tion potential, or indirectly through changes in subnuclear associations

A direct transcriptional effect could result from changes

in intramolecular interactions or altered post-translational modifications The first would fit with the finding of a main conjugation site (K527) within the previously identified EVES region of c-Myb [51] However, in our hands the reported EVES–DBD interaction, when assayed in a Gal4-two hybrid system, is rather weak and technically not suitable to investigate whether it is modulated by sumoy-lation We did test the other possibility of altered post-translational modifications in experiments where we compared c-Myb wild type and the 2KR mutant with respect to CBP interaction (CoIP experiments) and level of acetylation, but did not observe any differences related to the mutation (data not shown) Other possible direct mechanisms exist: sumoylation could affect transcription

as an intrinsic part of the transcriptional activation process through interference with ubiquitylation Recent reports have shown an unexpected involvement of ubiquitylation in transcriptional activation [52–54] Bies et al [17] propose that sumoylation stabilizes the c-Myb protein This cannot, however, explain the increased activity of 2KR, as it was reported that mutating the sumoylation sites has no effect

on the stability of the protein [17]

Another possibility is that SUMO-1 could mediate protein–protein interactions, making the sumoylated pro-tein able to interact with other propro-teins than the nonsumoy-lated protein, which is the case for PML [28,43] This concept that SUMO-1 conjugation stabilizes higher order protein complexes was recently suggested as a common theme for sumoylated proteins [18] We therefore examined whether PIASy-enhanced sumoylation of c-Myb would alter its distribution within the nucleus We observed that the portion of c-Myb in the insoluble part of the nucleus was increased after cotransfection with PIASy in a nuclear

Trang 9

matrix preparation experiment (Fig 6) This experiment

also showed that there is an accumulation of sumoylated

Myb in the insoluble fraction of the nucleus, indicating that

sumoylation stabilizes the association of c-Myb with

insoluble structures in the nucleus The mechanism for this

altered distribution remains to be elucidated We were not

able to detect direct interactions between c-Myb and PIASy

in cotransfection experiments (results not shown) suggesting

that it is not simply PIASy that sequesters c-Myb into the

nuclear matrix Whatever the mechanism, it is possible that

the trafficking of 2KR changes compared to wild type

c-Myb, and this leads to subtle changes in localization or

subnuclear associations This might cause secondary effects

resulting in the observed increased activity of 2KR The

emerging picture of the functional nuclear architecture

consisting of specialized domains with distinct biological

functions implies that most nuclear proteins are regulated

by and exert their functions from higher order protein

complexes at specific locations [55,56] If, as suggested here,

sumoylation is involved in regulating the association of

c-Myb with higher order complexes, it would be important

to study the effects of sumoylation of c-Myb in a more

biological context than transfected reporter assays provide

Deletion of the carboxy-terminal region of c-Myb

augments its transcriptional and transformation properties

(reviewed in [1,8,57]) For this reason the carboxy-terminal

part of the protein has been referred to as a negative

regulatory domain (NRD) More detailed mapping

sugges-ted the presence of two subdomains each contributing to the

NRD effect, the first of which harbours a putative leucine

zipper domain [58] The second subdomain spans the amino

acid residues 495–640 in chicken c-Myb [59,60], and thus

encompasses both sumoylation sites It has been proposed

that an additional cellular protein is required for negative

regulation of transcriptional activation by the NRD [8]

NRD regions in several transcription factors, including

c-Myb, share a common motif called the SC motif (synergy

control) [44], which appears to limit the transcriptional

synergy of these regulators through a mechanism involving

altered higher-order protein–protein interactions It is

intriguing that this motif matches exactly the consensus

sequence for SUMO-1 conjugation, and several of the

proteins previously identified to contain the SC motif have

later been shown to be sumoylated in these sites, for

example c-Myb [17], C/EBP [61], AR [47] and Sp3 [48] We

therefore propose that sumoylation of the NRD region of

c-Myb makes an important contribution to its negative

influence on transactivation properties This effect may

rely on alterations in higher-order interactions with

cooper-ating proteins or subnuclear structures Sumoylation as an

effector of NRD function may thus be a working model

linking effects on transcriptional activation with effects on

subnuclear associations

Acknowledgements

This work was supported by The Norwegian Research Council (ØD,

TØA, ON, OB, OSG), The Norwegian Cancer Society (OB, OSG), and

the Anders Jahres Foundation (OSG) We thank Tone Berge for

construction of plasmids expressing C-terminal tagged c-Myb We are

grateful to A Leutz for providing the HD11 cell line, and to Dr

Jonathan P Sleeman for the source of the 5E11 monoclonal antibody.

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