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Enforced expression of UXT in Evi1-expressing Rat1 fibroblasts suppresses cell transformation and UXT may therefore be a negative regulator of Evi1 biological activity.. Our results show

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EVI1 and suppresses cell transformation

Roger McGilvray1, Mark Walker2and Chris Bartholomew1

1 Department of Biological & Biomedical Sciences, Glasgow Caledonian University, UK

2 CRUK Beatson Laboratories, Beatson Institute for Cancer Research, Glasgow, UK

EVI1 is a member of the PR domain family of

pro-teins [1], which include PRDM1 (PRDI-BF1) [2],

PRDM2 (RIZ) [3] and PRDM16 (MEL1) [4],

and share structural similarities with N-terminal PR

domains, functional similarities with transcriptional

repressor activities, have multiple cys2⁄ his2 zinc-finger

motifs, and roles in cell differentiation and

tumorigen-esis The complex phenotype observed in Evi1

knock-out mice suggests that it has a role in a range of

biological processes including general cell proliferation,

vascularization and cell-specific developmental

signal-ling [5] The pleiotropic phenotype reflects Evi1’s

func-tions as a transcription factor [6] as well as its ability

to interfere with several signalling pathways through interactions with Smad [7,8] and JNK [9] proteins The Evi1 transcriptional repressor protein is essen-tial for normal development [5] and when inappropri-ately expressed participates in the progression of a subset of leukaemias and myelodysplasias [10] In vitro studies have shown that a number of biological prop-erties can be attributed to the Evi1 protein including: (a) deregulation of cell proliferation [11]; (b) inhibition

of transforming growth factor-b, bone morphogenic protein and activin signalling [7,8]; and (c) inhibition

of stress-induced apoptosis [9] These activities require the DNA-binding domains ZF1⁄ ZF2 [12,13] and a

Keywords

ART-27; cell transformation; EVI1; leukemia;

ubiquitously expressed transcript

Correspondence

C Bartholomew, Glasgow Caledonian

University, Department of Biological &

Biomedical Sciences, City Campus,

Cowcaddens Road, Glasgow G4 OBA, UK

Fax: +44 (0)141 331 3208

Tel: +44 (0)141 331 3213

E-mail: c.bartholomew@gcal.ac.uk

(Received 30 June 2006, revised 10 May

2007, accepted 8 June 2007)

doi:10.1111/j.1742-4658.2007.05928.x

The EVI1 transcriptional repressor is critical to the normal development of

a variety of tissues and participates in the progression of acute myeloid leu-kaemias The repressor domain (Rp) was used to screen an adult human kidney yeast two-hybrid library and a novel binding partner designated ubiquitously expressed transcript (UXT) was isolated Enforced expression

of UXT in Evi1-expressing Rat1 fibroblasts suppresses cell transformation and UXT may therefore be a negative regulator of Evi1 biological activity The Rp-binding site for UXT was determined and non-UXT-binding Evi1 mutants (Evi1D706–707) were developed which retain the ability to bind the corepressor mCtBP2 Evi1D706–707 transforms Rat1 fibroblasts, show-ing that the interaction is not essential for Evi1-mediated cell transforma-tion However, Evi1D706–707 produces an increased proportion of large colonies relative to wild-type, showing that endogenous UXT has an inhibi-tory effect on Evi1 biological activity Exogenous UXT still suppresses Evi1D706–707-mediated cell transformation, indicating that it inhibits cell proliferation and⁄ or survival by both Evi1-dependent and Evi1-independ-ent mechanisms These observations are consistEvi1-independ-ent with the growth-suppressive function attributed to UXT in human prostate cancer Our results show that UXT suppresses cell transformation and might mediate this function by interaction and inhibition of the biological activity of cell proliferation and survival stimulatory factors like Evi1

Abbreviations

CtBP, C-terminal binding protein; DBD, DNA-binding domain; GST, glutathione S-transferase; Rp, repressor domain; SD, synthetic dropout medium; UXT, ubiquitously expressed transcript.

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200-amino acid transcriptional repressor domain

(Rp) [6]

Rp is critical to the biological activity of Evi1 We,

and others, have shown that Rp binds the C-terminal

binding protein (CtBP) family of corepressor proteins

that interact via two conserved PLDLS-like motifs

[14,15] Critically, the ability of Evi1 to repress

tran-scription, deregulate cell growth [14] and inhibit

trans-forming growth factor-b, bone morphogenic protein

and activin signalling [7,8,15], all rely on the

recruit-ment of CtBP

Several lines of evidence suggest that Rp is involved

in additional protein interactions that may also

con-tribute to Evi1’s broad spectrum of biological

activit-ies C-terminal deletion mutants of Rp, which retain

CtBP-binding sites, create more effective repressors [6],

suggesting that this activity is regulated Naturally

occurring splice variants in both human and murine

Evi1 exist that insert nine amino acids (FP⁄

QLPDQRTW) into Rp [16,17], which may either

create or disrupt novel or pre-existing interactions,

respectively Inspection of aligned human and murine

Rp primary amino-acid sequences with the

correspond-ing region of the related MEL1 protein [4] reveal

signi-ficant stretches of conservation in addition to the

CtBP-binding sites, suggesting additional common

activities might have been conserved during evolution

for these two proteins

To date, several Evi1-binding proteins have been

identified using a candidate protein approach It

remains possible that Evi1 interacts with as yet unknown cellular proteins responsible for regulating biological activity To investigate this, we screened a yeast two-hybrid library to identify new Rp-binding partners

Results

Isolation of a new Evi1 Rp-domain binding protein

Rp was subcloned from pGBT9Rp [14] into the kana-mycin-selectable yeast vector pKGI [18] to create pKGIRp (see Experimental procedures) As expected, pKGIRp produces a GAL4 DNA-binding domain (DBD)⁄ Rp protein which interacts with mCtBP2 in yeast AH109 cells, confirming its suitability for use

in screening a yeast two-hybrid library (Fig 1A–C; pKGIRp, pGAD10mCtBP2)

In total, 1· 106 independent clones from a human adult kidney yeast two-hybrid library (Clontech) were screened with pKGIRp in AH109 cells Initially, 37 potentially interacting clones were identified of which only 17 grew under more stringent conditions Target plasmid DNAs recovered from these clones were intro-duced into AH109 cells with pKGIRp and growth was reassessed Six recombinant plasmids contained genes encoding putative Rp-interacting proteins (Table 1, secondary screen) Sequencing of the plasmid DNA inserts revealed that five were HuCtBP2 (A6, A7, A10,

Fig 1 Interaction of Rp and A1 in yeast

cells (A,D) Yeast AH109 cells (Clontech)

were transformed with the combination of

plasmids shown The growth of single yeast

colonies containing these plasmids are

shown in (B) and (E) on SD lacking L

-histi-dine and adenine and (C) on SD plus L

-histi-dine and adenine.

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A14 and A37) The remaining clone, A1, contained a

novel gene This gene was isolated repeatedly from

additional independent library screens (data not

shown) and together with HuCtBP2 represents the

only Rp-interacting proteins identified in the human

adult kidney library

AH109 cells only grow under stringent conditions

when they contain both Rp and A1 expressed as

GAL4DBD and GAL4AD fusion proteins, respectively

(Fig 1A–C; pKGIRp, pGAD10 A1), showing that

these proteins interact To see if A1 and Rp interact

irrespective of their fusion partners, GAL4AD or

GAL4DBD, a domain swap was undertaken A1 and

Rp were inserted into pGBT9 (pGBT9 A1) and

pGADT7 (pGADT7 Rp), respectively, and shown to

still interact in AH109 cells (Fig 1A–C; pGBT9 A1,

pGADT7 Rp) The yeast two-hybrid assay also

revealed that A1 can homodimerize in AH109 cells

(Fig 1D,E; pGBT9 A1, pGAD10 A1) Furthermore,

the interaction of A1 with Rp is specific because A1

does not interact with laminin, p53 (Fig 1A–C;

pGAD10 A1) or mCtBP2 (Fig 1D,E; pGBT9 A1⁄

pGAD10mCtBP2)

A1 is identical to a novel gene called UXT

The sequence of A1 is shown in Fig 2A It consists of

a 546-nucleotide cDNA, which includes a partial

poly(A) tail There is an unbroken reading frame of

162 amino acids that is continuous with the vector

GAL4DBD and terminates with TGA (Fig 2A) at

nucleotide 487 Inspection of the sequence shows that

the first ATG (Fig 2A) fits the Kozak consensus for

translation initiation [19], suggesting that the gene

nor-mally encodes a putative protein of 157 amino acids

with a predicted molecular mass of 18.2 kDa

Interro-gation of the NCBI nucleotide database revealed the

identity of A1 with a novel gene designated UXT (AF092737) that encodes a 157-amino-acid protein (Fig 2A) A1 is subsequently referred to as UXT The tissue distribution of UXT was examined using northern blot analysis This shows that UXT produces

an abundant transcript of 750 bp in all tissues exam-ined, with the highest expression levels in heart, skel-etal muscle, pancreas, peripheral blood leukocytes, thyroid and lymph node (Fig 2B) The transcript size

is consistent with A1 being almost full length, allowing for an additional 39 5¢ nucleotides described for UXT and a 200 nucleotide poly(A) tail

The tissue distribution of UXT shows that it is expressed in the same tissues as Evi1, including lung, kidney, ovary and heart RT-PCR was performed to confirm that both genes are expressed simultaneously

in the same cells Evi1 is abundantly expressed in the leukaemia cell line DA-3 [20] and primary mouse embryo fibroblasts (MEFS), but not in lymphoma-derived monocytic U937 cells (Fig 2C) UXT is expressed in all cell types examined (Fig 2C), confirm-ing that transcripts for both genes coexist in cells in which Evi1 is expressed, including leukaemia cells where Evi1 has been activated and in cells where Evi1

is normally expressed such as fibroblasts

UXT binds full-length Evi1 UXT⁄ Evi1 binding was confirmed using a glutathi-one S-transferase (GST)-pull down assay GST–Rp and GST–UXT fusion proteins were expressed and purified from Escherichia coli strain pLysS cells using bacterial expression vectors (see Experimental procedures)

35S-Methionine-labelled in vitro-translated UXT and Evi1 proteins were produced using the expression vectors pCDNA3–UXT (Experimental procedures) and pRC⁄ CMV FL [6], respectively (Fig 3A) GST pull-down assays were performed with combinations of

Table 1 Isolation of Rp-interacting proteins in yeast The number of colonies obtained (clone A1 to A37), their growth on various selective media (SM), and their production of a- and b-galactosidase from both the primary and secondary library screening are shown ND, not done.

A20, A24, A25, A26, A29, A32, A33

Growth on SM -Trp ⁄ -Leu ⁄ -His ⁄ -Ala A1, A6, A7, A10, A11, A14, A15, A17

A1, A6, A7, A10, A14, A37 A20, A24, A25, A26, A29, A32, A33 A36, A37

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Fig 2 (A) Sequence of clone A1 The

nuc-leotide sequence of clone A1 is shown.

Below is the sequence of UXT (lower case)

and regions of identity are indicated by -.

Below the nucleotide sequence is shown

the primary amino acid sequence using the

single letter code The bold nucleotide and

amino acid sequence is partial pGBT9 GAL4

DBD vector nucleotide and amino acid

sequence Bold boxed nucleotide sequences

show the predicted translation initiation site

for UXT ⁄ clone A1 and the translation

termin-ation site The boxed amino acid sequence

represents translation of predicted 5¢ UXT

noncoding leader sequence that maintains

the reading frame of GAL4 AD and UXT (B)

Northern blot analysis of UXT expression.

Human MTNTMblots (Human, Human II and

Human III; Clontech) containing 2 lg per

lane of the indicated poly(A + ) RNAs were

hybridized to a 32 P-labelled UXT (A1) probe.

Filters were washed stringently, 0.1· NaCl ⁄

Cit, 0.1% SDS, 65 C and bands were

visu-alized by autoradiography (C) RT-PCR

analy-sis of total cellular RNA derived from the

indicated cells using human ⁄ mouse-specific

Evi1 (HME1 ⁄ HME2), Uxt (HMUXT5 ⁄

HMUXT3) and Gapdh (GAPDH5¢ ⁄ GAPDH3¢)

primers The expected size fragments for all

three genes: Evi1 (467 bp); Uxt (278 bp) and

Gapdh (451 bp) are indicated by arrows.

M indicates the 1kb hyperladder (Bioline).

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bacterially expressed and 35S-methionine-labelled

pro-teins and the results are shown in Fig 3 These

con-firm that UXT binds Rp (Fig 3B, lane 5) and

furthermore also interacts with full-length Evi1

(Fig 3B, lane 7) Formation of the complexes are

spe-cific as they do not occur with GST alone (Fig 5B,

lanes 2 and 3)

The UXT⁄ Evi1 interaction was also confirmed in

mammalian cells using coimmunoprecipitation UXT

was inserted inframe with three copies of the HA

epi-tope tag into the expression vector pcDNA3 to create

pcDNA33HAUXT (Experimental procedures) Cells

from the human embryonic kidney cell line BOSC-23

were transiently transfected with various combinations

of the C-myc epitope-tagged Evi1-encoding vector

pcDNA3EVI1myc (A Coyle & C Bartholomew,

unpub-lished) and pcDNA33HAUXT, and portions of the

cell extracts were examined either directly by western

blot analysis or immunoprecipitated prior to western

blotting Western blot analysis of whole-cell extracts

with either HA-specific a-12CA5 or C-myc-specific a-9E10 shows the expected size epitope-tagged 21 kDa UXT (HAUXT) and 145 kDa Evi1 (EVI1myc) proteins, respectively, in cells transfected with the corresponding expression vectors (Fig 3C) Immuno-precipitation of cell extracts and western blot analysis with a-myc reveals EVI1myc in cells transfected with pcDNA3EVI1myc, as expected (Fig 3; IP a-9E10) In addition, western blot analysis of a-myc-immunopre-cipitated cell extracts with a-HA shows HAUXT only

in those extracts that also contain EVI1myc, confirm-ing that these two proteins form a complex Examina-tion of the UXT–Evi1 interacExamina-tion at the endogenous level using the same method must await new reagents

to be developed

UXT suppresses Evi1-mediated transformation Next we investigated whether UXT has an effect on Evi1 biological activity by examining the impact of

Fig 3 (A) SDS–PAGE of in vitro translated products showing 35 S-labelled UXT (lane 1) and Evi1 (lane 2) (B) Analysis of GST pull-down assays by SDS–PAGE White box indicates bacterially derived GST protein, stippled box represents in vitro translated UXT and bacterially derived GST–UXT fusion protein Grey box represents bacterially derived GST–Rp fusion protein Evi1 zinc-finger domains and repressor domains are indicated by black and grey boxes, respectively Hatched box shows acidic domain (C) Interaction of Evi1 and UXT (A1) in mammalian cells Various combinations of pcDNA3Evi1myc (4 lg) and pcDNA33HAUXT (1 lg), indicated by +, were transiently transfected into BOSC-23 cells as described previously [2] One-tenth of whole-cell extracts were utilized directly for western blot analysis and the remainder was immunoprecipitated with a-myc (Santa Cruz Biotechnology, IP a-9E10) prior to western blot analysis Whole-cell extracts and immunoprecipitation a-9E10 extracts were resolved by SDS–PAGE (10%) and sequentially probed with a-haemagglutinin (Boehringer Mann-heim, MannMann-heim, Germany; WB a-12CA5) and a-myc (WB a-9E10) mAb Proteins were visualized by ECLTM(Amersham Pharmacia Biotech) Protein size was estimated by comparison with Full Range Rainbow TM molecular mass markers (Amersham Pharmacia Biotech, not shown) Evi1myc and HAUXT fusion proteins are indicated by arrows.

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UXT expression on Rat1 and RatFL

(Evi1-trans-formed cells) cell transformation The retroviral

expression vector p50MHAUXTzeo, containing UXT

fused inframe to a HA tag, was created Recombinant

retrovirus produced in BOSC-23 cells was used to

infect Rat1 and RatFL cells and cell populations

expressing UXT were selected Neither UXT

expres-sion in Rat1 cells nor empty vector controls were

transforming (Fig 4A, lanes 1,3,4), whereas Evi1 was

(Fig 4A, lane 2) However, enforced expression of

UXT reduced the number of transformed colonies

pro-duced by RatFL cells by 50% (Fig 4A, lane 5) The

empty vector control had no effect in RatFL cells

(Fig 4A, lane 5) Cell extracts prepared from the var-ious cell populations confirmed that they each produce the expected UXT HA-tagged fusion protein (Fig 4B) The effect of UXT on Evi1 transcriptional repressor activity was also examined but no significant changes were observed (data not shown)

An Evi1 Rp domain mutant lacking UXT-binding activity enhances Rat1 transformation activity The UXT-binding region of Rp was determined using

a series of deletion mutants (Experimental procedures) using the yeast two-hybrid assay Binding was retained when the Rp fragment (514–724) was deleted from the N-terminus to amino acid 634 (fragment 634–724), but lost upon C-terminal deletions between 715 and 695 (data not shown) A series of refined deletion mutants were created from the C-terminus of the 634–715 frag-ment to determine the minimum deletion required to lose UXT-binding activity Yeast two-hybrid assays revealed that Rp UXT binding is lost upon deletion

of amino acids 706–707 (Fig 5A–C, quadrant 7–9) Western blot analysis with a-GAL4DBD confirmed expression of equally abundant correct size proteins (fragments were subcloned from pGBT9 to pGBKT7 for this purpose; data not shown)

The ability of an Rp-deletion mutant lacking only amino acids 706 and 707 (Rp D706–707) to bind UXT and mCtBP2 was examined using the yeast two-hybrid assay Results confirm that this mutant is unable to bind UXT (Fig 5D–F, quadrant 2) but retains the ability to bind mCtBP2 (Fig 5D–F, quadrant 5) The transforming activity of Evi1 containing the UXT-binding mutant Rp domain was investigated in Rat1 cells The Rp domain of the previously described vector, p50MRpWTneo, which has identical trans-forming activity to p50M4.6neo [14], was substituted for RpD706–707 to create p50MRpD706–707neo and populations of Rat1 cells expressing this gene were selected and tested for transformation using a soft agar colony assay The results show that Evi1D706–707 gen-erates the same number of transformed Rat1 cell col-onies as wild-type Evi1 (data not shown) However, the mutant protein produces a higher proportion of larger colonies (Fig 6A) Figure 6B shows the percent-age of total colonies that are > 0.3 mm in diameter, generated from populations of Rat1 cells expressing either WT or mutant forms of Evi1 Approximately 14% of colonies generated by Evi1D706–707 are

> 0.3 mm in size, whereas only 3–4% of Evi1 trans-formed colonies achieve these dimensions

To see if enforced expression of UXT inhibits the transforming activity of Evi1D706–707, the

colony-Fig 4 (A) Colony formation of Rat1 and RatFL cell populations

infected with the indicated retroviral vectors Numbers were

deter-mined for colonies > 0.1 mm derived from plating 103cells Error

bars indicate the average number of colonies observed from three

independent assays Schematic representation of Evi1 and UXT

proteins are as described in the legend to Fig 3 The HA epitope

tag is shown as a striped box (B) Western blot analysis with

a-haemagglutinin (as described in Fig 3) of whole-cell extracts

derived from Rat1 cells (1), p50MHAUXTzeo infected BOSC 23 (2),

Rat1 (3) and RatFL (4) cells The HAUXT fusion proteins are

indica-ted by an arrow.

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forming activity of Rat1 cells expressing both

exo-genous proteins was examined Populations of Rat1

cells infected with both p50MRpD706–707neo and

p50MHAUXTzeo were selected and the production of

macroscopic colonies in soft agar assessed The results

show that transformation is suppressed by  50% in

cells containing Evi1D706–707 and UXT (Fig 6C)

The empty vector control, p50MXZEO, has no effect

on Evi1D706–707 transforming activity

Discussion

We have shown that Evi1 can interact with at least

two proteins present in human adult kidney cells via

the Rp domain One interaction is with the human

homologue of mCtBP2, HuCtBP2, and its significance

has already been documented [14] Surprisingly, our

library screens did not isolate HuCtBP1, which

recog-nizes and binds the same conserved PLDLS motif as

HuCtBP2, despite PCR analysis of the human kidney

library demonstrating that HuCtBP1 was present (data

not shown) Evi1 has previously been shown to

associ-ate with both HuCtBP1 [21] and mCtBP1 (E Ritchie

and C Bartholomew, unpublished) The most likely

explanation is that no appropriate HuCtBP1 clones in

the library are expressed inframe with GAL4AD

Evi1’s interactions with UXT, an 18.2 kDa protein,

have not been described previously Enforced UXT

expression in RatFL cells moderately suppresses cell

transformation The Evi1 UXT-binding mutant

(Evi1D706–707) retains the ability to bind mCtBP2

and Rat1 cell-transformation activity is enhanced This

shows that UXT binding: (a) is not required for Evi1 interaction with mCtBP2; (b) is not required for Evi1-mediated cell transformation; and (c) has an inhibitory effect on Evi1 biological activity Interestingly, enforced expression of UXT with Evi1D706–707 in Rat1 cells still moderately suppresses cell transforma-tion The data suggest that at least part of the suppres-sor activity is mediated by its interaction with UXT but that enforced UXT expression has a general growth-suppressive activity that is independent of Evi1 In this regard, it would be interesting to see if UXT expression suppresses cell transformation by other oncogenes too

Recent studies suggest that Evi1 is a survival factor

It is able to protect cells against chemically induced apoptosis [22] and promote survival of haemopoietic stem cells [23] Therefore, negative regulation of Evi1 biological activity might compromise its survival func-tion, reducing cell transformation and⁄ or proliferation

by decreasing the ability of cells to proliferate

optimal-ly in new environments, for example, the anchorage-independent growth of Rat1 fibroblasts in soft agar displayed in the transformation assay

UXT is located on human chromosome Xp11 and was originally identified when searching for the X-linked genes responsible for Renpenning syndrome, Prieto syndrome and Sutherland–Haan syndrome that map to this region, although it does not appear to be involved in their development [24] As shown here, UXT is abundantly and ubiquitously expressed in all tissues examined Based on an EST database search it has been suggested that UXT is overexpressed in

Fig 5 Interaction of UXT deletion mutants with Rp and mCtBP2 in yeast cells (A,D) Yeast AH109 cells were transformed with the combination of plasmids shown The growth of single yeast colonies containing these plasmids are shown in (B) and (E) on

SD plus L -histidine and adenine and (C) and (F) on SD lacking L -histidine and adenine.

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tumour cells [24] and might have a role in

tumorigen-esis In this regard, it is interesting to note that acute

basophilic leukaemia is associated with translocation

t(X,6)(p11,q23) [25] and therefore UXT itself is a

can-didate for the disease-associated Xp11 gene or

alternat-ively its ubiquitously active promoter may deregulate

expression of a novel leukaemia gene located on 6q23

The function of UXT is not known, but it has

previ-ously been shown to associate with several other

pro-teins, each of which has a role in the regulation of

gene transcription These include the leucine-rich

pen-tatricopeptide repeat-motif-containing (LRPPRC)

pro-tein [26] and the transcriptional coactivator CITED2

[27] UXT is also known as ART-27, a transcriptional

coactivator that interacts with the N-terminus of the

androgen receptor [28] Interestingly, enforced

expres-sion of UXT⁄ ART-27 in LNCaP prostate cancer cells

inhibits proliferation and furthermore its expression is

downregulated in human prostate cancer [29] This

proliferation-suppressive activity is consistent with the

inhibition of Evi1-mediated Rat1 cell transformation

mediated by UXT in our studies Both these

experi-mental observations are in direct contradiction to the

earlier interpretation derived from interrogation of the

EST database [24] Furthermore, UXT expression has

also been shown to be elevated in bladder, breast,

ovary and thyroid tumours suggesting it may be a

tumour marker [30] This apparent discrepancy may be

reconciled if UXT effects on cell transformation are tissue specific or that either up- or downregulation contributes to oncogenesis Alternatively, there are at least two naturally occurring UXT splice variants, transcript variant 1 encoding a 157-amino acid protein (NM153477) and transcript variant 2 encoding an N-terminal truncated protein of 145 amino acids (NM004182), which might have opposing effects on cell transformation In this regard, it will be interesting

to investigate both the form of UXT and the structural integrity of the gene coding sequences in tumour cells where its expression is elevated

Several lines of evidence indicate that UXT regulates cell proliferation UXT is a target gene for the E2F family of transcription factors that regulate transition through the G1⁄ S phase boundary of the cell cycle [31] Both E2F1 and E2F6 inhibit UXT gene expres-sion [31,32] Furthermore, E2F6 corepresses UXT and other genes with common functions in tumour sup-pression, suggesting that it might have a similar activ-ity [32], consistent with its abilactiv-ity to inhibit cell transformation

UXT has also been isolated as STAP1 and classified

as a member of the a class prefoldin family [33] UXT (STAP1) is a component of a large protein complex that can regulate transcription in HeLa cells, consistent with its interaction with the Evi1 transcription factor observed here UXT has also been shown to be located

Fig 6 (A) Photograph of colonies formed

by Rat1 cell populations expressing the

indicated Evi1 proteins using a Leica GZ6

microscope (B) Histogram showing the

percentage of soft agar colonies > 0.3 mm

generated by populations of Rat1 cells

expressing either wild-type (Evi1) or mutant

(Evi1D706–707) Evi1 The percentage was

determined by counting total number of

col-onies generated and the number of colcol-onies

> 0.3 mm Colony numbers were

deter-mined from two independent experiments

(1 & 2) Control 1 and 2 are parental Rat1

cells Error bars indicate variation between

three independent assays for each

experi-ment (C) Histogram showing total number

of soft agar colonies > 0.3 mm produced

per 1000 Rat1 cells expressing the indicated

proteins Control is empty p50MXZEO

retro-viral vector Error bars indicate the average

number of colonies observed from three

independent assays No colonies are

observed in Rat1 cells in the absence of

Evi1 (not shown).

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in centrosomes and its overexpression disrupts

centro-some structure in human U2OS cells [30] It has been

suggested that UXT abnormality may cause

dysfunc-tion of the centrosome contributing to malignant

transformation [30] Centrosomes play a key role in

cell proliferation, serving both to nucleate polarized

microtubule arrays for mitotic spindle organization

and cytokinesis and providing a multiplatform scaffold

with protein-docking sites for integrating cellular

regu-latory events [34] This raises the possibility that Evi1

might contribute to the regulation of cell proliferation

by interacting with a component of centrosomes

Our results show that UXT inhibits Evi1-mediated

cell transformation in addition to the previously

des-cribed inhibition of cell-proliferation activity and

therefore may be a negative regulator of cell growth

UXT interacts with Evi1 and may be a direct negative

regulator of its biological activity The precise

molecu-lar mechanism by which UXT reduces Evi1 activity is

unknown UXT might mediate its negative control of

cell proliferation and transformation by directly

inter-acting with and regulating the activity of factors that

stimulate this biological activity such as Evi1 In

addition, UXT mediates its negative activity on cell

proliferation and transformation indirectly, by an

independent mechanism

Experimental procedures

Construction of plasmids

pKGIRp was created by inserting a EcoRI⁄ BamHI Rp

domain fragment from pGBT9Rp [14] into pKG1 [18]

pGBT9A1 was created by excising A1 as a BglII fragment

from pGAD10 A1and inserting into a BamHI site of

pGBT9 pGADT7Rp was created by excising a EcoRI⁄

BamHI Rp fragment from pKG1Rp and inserting into the

corresponding site of pGADT7 (Clontech, Mountain View,

CA) pGEXUXT was created by PCR amplification with

5¢- and 3¢-oligonucleotides CGCTGGATCCCGGGAGG

AGCCCATCATG and GGAAGAATTCTCAAATTCCA

GGAAAAAACCA, respectively, and insertion of UXT

fragments into BamHI⁄ EcoRI of pGEX2 (Promega,

Madi-son, WI) pGEXRp was created by PCR amplification with

5¢- and 3¢-oligonucleotides AAGCGGATCCCGCATTCT

CTCAATCAATG and AAGCTGAATTCGTAGCGCTC

TTTCCCCT and insertion of Rp fragments into BamHI⁄

EcoRI of pGEX1 (Promega) pcDNA3UXT was created by

inserting a HindIII⁄ BamHI UXT PCR fragment amplified

from pGAD10 A1 with oligonucleotides AATTCAA

GCTTGCGCAATGAAGGTGAAGG and AATTCGGAT

CCTCAATGGTGAGGCTTCTC pcDNA33HAUXT was

created by simultaneous ligation of a NcoI⁄ NotI-digested

UXT PCR fragment, amplified from pGAD10 A1 with 5¢- and 3¢-oligonucleotides GAATCCATGGCGACGAC GCCCCCTAAGCG and GAATTGCGGCCGCCTCAAT GTGAGGCTTC, respectively, with a NcoI⁄ EcoRI frag-ment containing three copies of the HA epitope from S3H-ERK2 (gift from W Kolch, Beatson Institute, Glasgow, UK) and EcoRI⁄ NotI-digested pcDNA3 (Invitrogen, Carls-bad, CA) p50MHAUXTzeo was created by PCR amplifi-cation of pGAD10 A1 with 5¢- and 3¢-oligonucleotides AGCTTGCGGCCGCATCATGTACCCATACGATGTTC CAGATTACGCTGCGACCCCCCTAAGCG and GCTG AATTCTCAATGGTGAGGCTTC which was digested with NotI⁄ EcoRI and inserted into the corresponding site

of p50Mxzeo [14] Rp domain deletion mutants were cre-ated by inserting EcoRI⁄ NotI-digested PCR fragments gen-erated using the following 5¢-oligonucleotide (E634)

3¢-oligonucleotides (N724) AATTGCGGCCGCTCAGTA GCGCTCTTTCCCCTT (N715) AATTGCGGCCGCTCA GTTCTCTGGCAGGGTGTT or EcoRI⁄ BamHI-digested PCR fragments generated with 3¢-oligonucleotides (B713) AGCTTGGATCCTATGGCAGGGTGTTGGGAGGAGC, (B711) AGCTTGGATCCTAGGTGTTGGGAGGAGCTC

GGAAGCTGAA (B707) AGCTTGGATCCTAAGCTCG GAAGCTGAACATGGA (B705) AGCTTGGATCCTA GAAGCTGAACATGGAGGGCAC, into EcoRI⁄ NotI-digested pGBT9N [14] or EcoRI⁄ BamHI-digested pGBT9 pGBT9RpD706–707 was created by site-directed mutagen-esis (QuickChange XL system, Stratagene, La Jolla, CA) of pGBT9Rp [14] with oligonucleotides 5¢-CCCTCCATGTT

GGTGTTGGGAGGGAAGCTGAACATGGAGGG The same primers were used for site directed mutagenesis of p50MRpWTneo [14] to create p50MRpWTD706–707neo

Cell culture, transfections, CAT and b-galactosidase assays

RatFL cells have been described previously [11] Rat1,

Rat-FL, Bosc-23, HEK293 and primary mouse embryo fibro-blasts were all maintained in high glucose Dulbecco’s modified Eagle’s medium supplemented with 10% fetal bovine serum, sodium pyruvate and glutamine U937 cells were maintained in RPMI-1640 supplemented with 10% heat-inactivated fetal bovine serum, sodium pyruvate and glutamine DA-3 cells were similarly maintained in the pres-ence of 10% WEHI-3-conditioned medium Procedures for transfections, production of helper-free recombinant retrovi-rus, retroviral infections and growth in soft agar, CAT and b-galactosidase assays have all been described previously [6] Cells infected with zeocin containing retroviral vectors were selected and maintained in 1 mgÆmL)1 zeocinTM (Invitro-gen)

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Yeast two-hybrid assay

For the primary screen, AH109 competent cells were

pre-pared and transformed as described by the suppliers

(Clontech) with 1 mg pKGIRp and 0.8 mg human kidney

cDNA plasmid library (Clontech) Total numbers of

trans-formed colonies were estimated by growing an aliquot of

transformed cells for 3 days, at 30C on synthetic dropout

medium (SD), 0.67% yeast nitrogen base without amino

acids (Difco Laboratories, Sparks, MD), 0.06%

CSM-HIS-LEU-TRP (Bio101, Inc., Irvine, CA), 2% glucose, pH 5.8,

1.5% select agar (Life Technologies, Grand Island, NY)

and 20 lgÆmL)1 l-histidine HCl (Sigma, St Louis, MO;

H-9511) The remaining cells were grown for 14 days on

SD without l-histidine Growth of any colonies was

subse-quently examined under stringent conditions on SD lacking

both l-histidine and adenine by substituting CSM for DO

supplement (-ADE,-HIS,-LEU,-TRP; Clontech)

Plasmid DNA was isolated from yeast colonies by

scraping into 1 mL TE buffer, pelleting cells and

resus-pending them in 0.5 mL 10 mm K2HPO4 pH 7.2, 10 mm

EDTA, 50 mm 2-mercaptoethanol, 0.25 mgÆmL)1

zymo-lase, at 37C for 30 min, mixing with 0.1 mL 25 mm

Tris⁄ HCl pH 7.5, 25 mm EDTA, 2.5% SDS, at 65 C for

30 min followed by 10 min at 0C with 166 lL 3 m

KOAc E coli strain DH5a electroporation-competent

cells were prepared according to Sambrook et al [35],

electroporated as described by Clontech and transformed

colonies selected on Luria–Bertani plates containing

50 lgÆmL)1 ampicillin Plasmid DNAs were prepared

using a NucleoSpin plus miniprep plasmid extraction kit

(Clontech) a- and b-galactosidase assays, respectively,

were performed as described by Clontech

GST pull-down assay

Bacterial cultures containing pGEX expression vectors were

induced with 1 mm isopropylthio-b-d-galactoside for 3 h

and cells were resuspended and sonicated in NETN buffer

(20 mm Tris pH 8.0, 100 mm NaCl, 1 mm EDTA, 0.5%

NP40) Extracts were cleared in a microfuge at 4C and

GST-fusion proteins bound to glutathione Sepharose by

mixing with glutathione Sepharose slurry equilibrated with

NETN at 20C for 30 min followed by centrifugation and

washing three times in NETN.35S-Labelled in vitro

transla-ted proteins were produced using TNT-coupled reticulocyte

lysates (Promega) GST pull-down assays were performed

by incubating GST-fusion protein⁄ glutathione Sepharose

conjugate (5 mg), 100 mg E coli cell extract and in vitro

translated protein in NETN, 4C, overnight Extracts were

washed three times in excess NETN, 1· in excess MTPBS

(150 mm NaCl, 16 mm Na2HPO4, 4 mm NaH2PO4, pH 7.3)

and bound protein eluted from complex in 50 mm

Tris⁄ 5 mm reduced glutathione

Immunoprecipitation

Cells were scraped into 0.25 mL of immunoprecipitation buffer [36], rapidly frozen, thawed at 0C for 1 h, then microfuged at 11 000 g for 10 min at 4C Supernatant was removed, and 25 lL was aliquoted as whole-cell extract for western blotting and the remainder incubated o⁄ n with anti-(c-myc a-9E10) serum (Santa Cruz Biotechnology, Santa Cruz, CA) at 4C and subsequently incubated with

50 lL of 50% slurry of rabbit anti-(mouse IgG)-coated protein A Sepharose beads for 2 h at 4C Beads were washed three times in immunoprecipitation buffer and pre-pared for western blot analysis

RT-PCR

Total RNA (0.2 lg, prepared using the RNazolTMB method) was amplified using the CalypsoTM RT-PCR system (BioGene Ltd., Kimbolton, UK) according to the manufac-turers instructions The coupled reaction was performed in a

MJ Scientific thermal cycler at 50C for 30 min, followed by amplification by 30 cycles of 30 s at 94C, 30 s at 55 C,

1 min at 72C, and a final 10 min at 72 C extension time using the following human⁄ mouse-specific primers: HME1 CCAGATGTCACATGACAGTGGAAAGCACTA; HME2 CCGGGTTGGCATGACTCATATTAACCATGG; UXT 5¢-GACAAGGTATATGAGCAGCTG; UXT 3¢-TTG ATATTCATGGAGTCCTTG; Gapdh5 ACCACAGTCCA TGCCATCAC; Gapdh3 TCCACCACCCTGTTGCTGTA PCR products were resolved by agarose gel electrophoresis (NuSieve GTG agarose, FMC)

Sequencing

A Licor automated sequencer was used for sequence determ\ination using SequiTherm EXCELII (Cambio, Cambridge, UK) and appropriate IRD-800 labelled primers

Site-directed mutagenesis

The QuickChange XL system (Stratagene) was used accord-ing to the manufacturer’s instructions

Western blot analysis

Whole-cell extracts were prepared as described previously [14] Proteins were examined by SDS⁄ PAGE, transferred to HybondTM-ECL nitrocellulose, incubated with appropriate antibodies and visualized with an ECL western blotting detection system (Amersham Pharmacia Biotech, Piscata-way, NJ) Protein sizes were estimated by comparison with Full Range RainbowTM molecular mass markers (Amer-sham Pharmacia Biotech)

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