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Intracellular localization and transcriptional regulation of tumornecrosis factor TNF receptor-associated factor 4 TRAF4 Heike Glauner1, Daniela Siegmund1, Hassan Motejadded2, Peter Sche

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Intracellular localization and transcriptional regulation of tumor

necrosis factor (TNF) receptor-associated factor 4 (TRAF4)

Heike Glauner1, Daniela Siegmund1, Hassan Motejadded2, Peter Scheurich1, Frank Henkler2,

Ottmar Janssen3and Harald Wajant1

1

Institute of Cell Biology and Immunology and2Institute of Industrial Genetics, University of Stuttgart, Germany;3Institute of Immunology, Christian-Albrechts-University of Kiel, Germany

To gain insight in the subcellular localization of tumor

necrosis factor receptor-associated factor (TRAF4) we

analyzed GFP chimeras of full-length TRAF4 and various

deletion mutants derived thereof While TRAF4–GFP (T4–

GFP) was clearlylocalized in the cytoplasm, the N-terminal

deletion mutant, T4(259–470), comprising the TRAF

domain of the molecule, and a C-terminal deletion mutant

consisting mainlyof the RING and zinc finger domains of

TRAF4 were both localized predominantlyto the nucleus

Passive nuclear localization of T4(259–470) can be ruled out

as the TRAF domain of TRAF4 was sufficient to form high

molecular weight complexes T4(259–470) recruited

full-length TRAF4 into the nucleus whereas TRAF4 was unable

to change the nuclear localization of T4(259–470) Thus, it

seems that individual T4(259–470) mutant molecules are

sufficient to direct the respective TRAF4–T4(259–470)

heteromeric complexes into the nucleus In cells forming

cell–cell contacts, TRAF4 was recruited to the sites of

con-tact via its C-TRAF domain The expression of some TRAF proteins is regulated bythe NF-jB pathway Thus, we investigated whether this pathwayis also involved in the regulation of the TRAF4 gene Indeed, in primaryT-cells and Jurkat cells stimulated with the NF-jB inducers TNF or phorbol 12-myristate 13-acetate (PMA), TRAF4-mRNA was rapidlyup-regulated In Jurkat T-cells deficient for I-jB kinase c (IKKc, also known as NEMO), an essential com-ponent of the NF-jB-inducing–IKK complex, induction of TRAF4 was completelyinhibited In cells deficient for RIP (receptor interactive protein), an essential signaling inter-mediate of TNF-dependent NF-jB activation, TNF-, but not PMA-induced up-regulation of TRAF4 was blocked These data suggest that activation of the NF-jB pathway is involved in up-regulation of TRAF4 in T-cells

Keywords: IKKc; NF-jB; T-cells; localization; TRAF4

The tumor necrosis factor (TNF) receptor-associated factor

(TRAF) familycomprises a group of adaptor proteins that

are involved in signal transduction bymembers of the TNF

receptor and IL1/Toll-receptor family[1,2] The TRAF

proteins are characterized bya C-terminal homology

domain of about 200 amino acids, called the TRAF

domain The TRAF domain mediates homo- and

hetero-merization of TRAF proteins and is also responsible for the

majorityof protein–protein interactions that have been

described for these molecules [1,2] The TRAF domain can

be subdivided into the highlyconserved carboxy-terminal

TRAF-C subdomain, consisting of an eight-stranded

anti-parallel b-sandwich structure and a less conserved

amino-terminal part, the TRAF-N domain, which is organized as a

coiled-coil [1,2] The TRAF domains form trimeric trefoil-like structures, with the three TRAF-C domains as leaves and the trimerized TRAF-N domains as the stalk [3–5] In mammalians six different TRAF proteins, designated as TRAF1–TRAF6, have been described With respect to the architecture of the N-terminal domain, TRAF1 is clearly distinct from all other TRAFs While the N-terminus of TRAF2–TRAF6 contains a highlyconserved RING domain followed bya regularlyspaced cluster of five or seven zinc fingers, the TRAF1 N-terminus onlycontains a single zinc finger and no obvious additional structural elements [1,2] While TRAF1–TRAF5 have been implicated mainlyin signaling bymembers of the TNF receptor family, TRAF6 primarilytransduces signals initiated byIL1/Toll receptors In particular, TRAF4 has been shown to interact with the lymphotoxin-b receptor and the p75 nerve growth factor receptor in in vitro binding assays [6,7] but the physiological relevance of these interactions remains to be elucidated While there is ample experimental evidence, including the analyses of knockout mice, for an important role of TRAF2, TRAF5 and TRAF6 in TNF receptor and IL1/Toll-receptor induced activation of NF-jB and JNK (c-Jun N-terminal kinase) [1,2], the role of TRAF1 and TRAF3 for signal transduction byTNF receptors is poorly understood In fact, B-cells from mice deficient in TRAF3 have a defect in immunoglobulin isotype switching in response to thymus-dependent antigens [8] and TRAF1 knockout mice show an increased TNF-R2-dependent

Correspondence to H Wajant, Institute of Cell Biologyand

Immunology, University of Stuttgart, Allmandring 31,

70569 Stuttgart, Germany.

Fax: + 49 711 685 7484, Tel.: + 49 711 685 7446,

E-mail: harald.wajant@po.uni-stuttgart.de

Abbreviations: CHX, cycloheximide; FLIP, fluorescence loss in

photobleaching; IKK, I-jB kinase; NF-jB, nuclear factor jB;

PBMNC, mononuclear cells; PMA, phorbol 12-myristate 13-acetate;

RPA, RNAse protection assay; TNF, tumor necrosis factor;

TRAF, TNF receptor-associated factor.

(Received 24 April 2002, revised 10 July2002,

accepted 13 August 2002)

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proliferation of CD8+ T-cells [9], but the molecular basis

of these defects has not been identified

TRAF4 is the most conserved phylogenetically, but also

the most distinct member of the TRAF family[1] Indeed,

the overall sequence identitybetween human TRAF4 and

its counterpart in Drosophila melanogaster (DmTRAF1) is

45%, whereas the closest related human TRAF shares only

26% sequence identity[1] In addition, expression of

TRAF4 and DmTRAF1 can be detected throughout

embryogenesis and is predominantly found in

undifferen-tiated cells, e.g neuronal precursors or epithelial progenitor

cells [7,10,11] Thus, it seems possible that DmTRAF1 and

mammalian TRAF4 represent conserved members of the

TRAF familywith related functions in differentiation of

vertebrate and invertebrate cells According to the broad

expression of TRAF4 in developing epithelial and neuronal

tissue, the analysis of TRAF4-deficient mice revealed a

neural tube closure defect as well as malformation of rib,

sternum, the spinal column and the upper respiratorytract,

the latter associated with an increase in pulmonary

inflammation [12,13] TRAF4 was cloned originallyin a

differential expression screen from a cDNA libraryof breast

cancer-derived metastatic lymph nodes and was found to be

located in the nucleus [14] However, another study, using a

different antibody, failed to detect TRAF4 in breast

carcinomas and reported a cytosolic localization of the

protein [7]

In this studywe found that deletion of the zinc finger

domain of TRAF4 results in nuclear localization without

disturbing the oligomerization status of the molecule This

opens the possibilitythat a zinc finger-dependent

mechan-ism retains TRAF4 in the cytoplasm and could provide an

explanation for the conflicting reports on the subcellular

localizations of TRAF4 TRAF4 is also recruited to sites of

cell–cell contacts under critical involvement of its C-TRAF

domain Finally, we show that TRAF4 is induced in T-cells

byTNF and treatment with phorbol ester under critical

involvement of I-jB kinase c (IKKc, also known as

NEMO), an essential component of the NF-jB signaling

pathway

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

Cells and reagents

The human cervical carcinoma cell line HeLa, the human

embryonic kidney cell line 293, the human breast cancer

cell line MCF-7 and the human epidermal carcinoma cell

line A431 were obtained from the American Type Culture

Collection (Rockville, MD, USA) The IKKc-deficient

Jurkat cell line and respective control cells were a gift

from S.-C Sun (Pennsylvania State University, USA) and

are described elsewhere [11] The RIP-deficient Jurkat

T-cell line and the corresponding parental Jurkat clone

were a gift from B Seed (Massachusetts General Hospital,

USA) and were described byTing et al [12] Cells were

maintained in RPMI medium (Biochrom, Berlin,

Germany) supplemented with 5% (HeLa and HEK293

cells) or 10% (Jurkat cells) fetal bovine serum Chemicals

and secondaryantibodies were obtained from Sigma

(Deisenhofen, Germany) The polyclonal TRAF4-specific

IgG preparation (C-20) was from Santa Cruz (Heidelberg,

Germany)

Plasmids

To construct human TRAF4 and TRAF4 deletion mutants (75–470, 259–470, 1–268, 1–307, 259–307, 259– 387) with carboxyl-terminal GFP or YFP tags, TRAF4 cDNA fragments with 5¢-end BamH1 overhangs and 3¢-end Sac2 overhangs were generated byproofreading PCR and HeLa cDNA as template The BamH1/Sac2 digested amplicons were ligated into the pEGFP-N1 and pEYFP-N1 vectors digested with Bgl2 and Sac2 To construct a deletion mutant consisting solelyof the C-TRAF domain

of TRAF4, an appropriate cDNA fragment of TRAF4 with a 5¢-end BamH1 overhang and 3¢-end Sac2 overhang was generated byproofreading PCR and inserted into Bgl2/Sac2 digested pEYFP-N1 vector (Clontech) To obtain non-GFP/YFP tagged TRAF4 expression con-structs, the GFP/YFP encoding cDNA stretch was removed from the corresponding GFP/YFP expression construct by Sac2/Not1 digest and subsequent religation of the blunt-ended vector–TRAF4 fragment GFP/YFP chi-meras of TRAF1, TRAF2 and TRAF3 were prepared in a similar way In case of TRAF3 a splice form was used lacking exons 7–10

Purification and stimulation of primary human T-lymphocytes

Mononuclear cells (PBMNC) were isolated from periph-eral blood of healthydonors byFicoll densitycentrifuga-tion The resulting PBMNC were then incubated with neuraminidase-treated sheep red blood cells at a ratio of

30· 106 PBMNC per ml sheep erythrocytes (10% sus-pension in RPMI) The mixture was separated bytwo rounds of Ficoll densitycentrifugation After the first gradient, the interphase containing nonrosetting cells was aspirated and the pellet with rosetted T-cells was carefully resuspended and centrifuged on the second gradient After aspirating the second interphase and ficoll, the sheep red blood cells were lyzed with ammonium chloride solution (Sigma, Deisenhofen, Germany) and the T-cells were washed twice with RPMI containing 10% fetal bovine serum The resulting cell population consisted of highly purified CD3+ T-cells (with an average percentage of CD3+ cells of 94–97%) The cells were kept overnight at

4C and then stimulated in 6 well plates for the indicated times at a densityof 30–50· 106 cells with or without PMA (5 ngÆmL)1, Sigma) and ionomycin (500 ngÆmL)1, Calbiochem) in 5 mL of RPMI/10% fetal bovine serum Finally, cells were carefully resuspended, washed once with NaCl/Pi, pelleted and stored at )20 C until RNA preparation As a control, untreated T-cells were collec-ted, washed and stored the same way For stimulation periods of 2, 4, 6 and 8 days, additional culture medium (2.5 mL each) was added after the second and fourth day T-cell blasts were generated by phytohaemag-glutinin (0.5 lgÆmL)1) stimulation of freshlyisolated PBMNCs for 3 days The cells were expanded with IL2-containing medium (10 UÆmL)1) for 3–5 days Dead cells were removed byFicoll densitygradient centrifugation and living cells were further expanded with IL2-supple-mented medium At the dayof restimulation (usuallyday 12–15) the population consisted of above 95% CD3+ T-cells

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RNAse protection assay (RPA) analysis

Cells were treated as indicated and total RNAs were isolated

with peqGOLD RNAPure (PeqLab Biotechnologie

GmbH, Erlangen, Germany) according to the

manufac-turer’s recommendations To detect transcripts for xIAP,

TRAF1, TRAF2, TRAF4, NAIP, cIAP2, cIAP1, TRPM2,

TRAF3, L32 and GAPDH total RNAs were analyzed

using a customer Multi-Probe template set (PharMingen,

Hamburg, Germany) Probe synthesis, hybridization and

RNase treatment were performed with the RiboQuant

Multi-Probe RNase Protection AssaySystem (PharMingen,

Hamburg, Germany) according to the manufacturer’s

recommendations After RNase treatment the protected

transcripts were resolved byelectrophoresis on a denaturing

polyacrylamide gel (5%) and analyzed on a

Phosphor-Imager with theIMAGEQUANTsoftware

Gelfiltration, subcellular, fractionation and Western

blotting

HEK293 cells (20· 106cells per mL) were electroporated

(4 mm cuvette, 250 V, 1800 lF, maximal resistance) in

medium with 5% fetal bovine serum, seeded on to two

150 mm tissue culture plastic plates and expanded for two days Cells were scraped with a rubber policeman into the medium, centrifuged (500 g, 5 min) and washed with medium The pellet was resupended in 300 lL of ice-cold

10 mMHepes, 10 mMKCl, 0.1 mMEGTA, 0.1 mMEDTA,

pH 7.9 All the following procedures were performed on ice

Fig 2 Subcellular localization of TRAF4 deletion mutants in isolated single cells HeLa cells were seeded on glass cover slides and transiently transfected with expression constructs for the indicated proteins Iso-lated single growing cells were selected for photography16–36 h after transfection.

Fig 1 Structure of fusion proteins used in this study The C-TRAF

domain (CTD) is shown in black and the N-TRAF domain (NTD) in

gray An open box denotes a zinc finger structure (Zn) and a RING

domain is represented bya pale graybox YFP or GFP are labeled The

amino acids are numbered according to the human TRAF sequences

available on GenBank (accession numbers U19261 (TRAF1), U12597

(TRAF2), U19260 (TRAF3) and X80200 (TRAF4).

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or at 4C For cell lysis 1/10 volumes of a protease inhibitor

cocktail (Boehringer Mannheim, Germany) and

Nonidet-P40 to a final concentration of 0.6% were added After

30 min on ice, the lysates were centrifuged at 10 000 g for

10 min and the supernatants were further cleared by

centrifugation at 50 000 r.p.m for 1 h in a TL-100 rotor

(Beckman, Munich, Germany) The S-100 supernatants

(250 lL) were then separated bysize exclusion

chromato-graphyon a Superdex 200 HR10/30 column (Pharmacia,

Freiburg, Germany) in 10 mMHepes, 10 mMKCl, 0.1 mM

EGTA, 0.1 mMEDTA, pH 7.9 with 0.5 mLÆmin)1

Sam-ples were collected in fractions of 0.5 mL and analyzed by

immunoblotting For calibration of the column

thyroglo-bulin (669 kDa), apoferritin (443 kDa), alcohol

dehydro-genase (150 kDa), bovine serum albumin (66 kDa),

carbonic anhydrase (29 kDa) and cytochrome c

(12.4 kDa), all purchased from Sigma (Deisenhofen,

Germany) were used For Western blot analysis 250 lL of

each fraction was precipitated with trichloroacetic acid and

dissolved in 60 lL of sample buffer Identical volumes

(30 lL) of the precipitated gel filtration fractions were

separated bySDS/PAGE and transferred to nitrocellulose

The GFP and the various TRAF4–GFP/YFP fusion

proteins were detected with 1 lgÆmL)1 of a mixture of

GFP-specific monoclonal antibodies (Roche, Mannheim,

Germany) and alkaline phosphatase-labelled goat

anti-(mouse IgG) (1 : 10000 dilution) For subcellular fraction-ation, cells were washed twice with NaCl/Piand half of the cells were used for preparation of cytoplasmic and nuclear extracts, respectively For preparation of cytoplasmic extracts, the cells were resuspended in 10 mM Tris/HCl,

2 mMMgCl2, pH 7.6 supplemented with protease inhibitors and incubated for 5 min on ice Then Triton X-100 was added to a final concentration of 0.5% After an additional

5 min cells were pressed 3–5 times through a 22-needle After centrifugation for 10 min at 14 000 g the supernatant was used for analysis For preparation of nuclear extracts, cells were treated as above, however, this time cells were onlycentrifuged for 10 min at 200 g The pellet was washed twice in 10 mM Tris/HCl, 2 mM MgCl2, pH 7.6 and resuspended in 20 mM Hepes, 1.5 mM MgCl2, 420 mM KCl, 1 mM EDTA, 25% glycerol, pH 7.9, supplemented with protease inhibitors The suspension was shaken gently for 30 min at 4C and finallynuclear lysates were obtained byremoval of insoluble material bycentrifugation for

15 min at 14 000 g at 4C

Immunofluorescence and confocal microscopy HeLa cells were seeded overnight on to 18 mm glass coverslips in square Petri dishes with 25 compartments The following day, cells were transfected with the indicated expression plasmid using Superfect reagent (Qiagen, Hilden, Germany) according to the manufacturer’s instructions For microscopic analysis transfected cells were fixed in 35% paraformaldehyde For bleaching experiments, cells were transfected in glass bottom dishes (MatTek Corporation, Ashland, MA, USA) and maintained during the experiment

in a conditioned chamber (37C, 5% CO2) for up to 2 h on

Fig 3 Exon–intron structure of human TRAF genes and Western blot

analysis of endogenous TRAF4 (A) The exon–intron structures of the

human genes encoding TRAF2 to TRAF6 according to the NCBI

entries NT_025667 (TRAF2), NT_010019 (TRAF3), NT_030828

(TRAF4), NT_021877 (TRAF5) and NT_024229 (TRAF6) were

mat-ched with the cDNA sequences encoding TRAF2 to TRAF6 [accession

numbers U12597 (TRAF2), U19260 (TRAF3), X80200 (TRAF4),

AB000509 (TRAF5) and U78798 (TRAF6)] Onlyexons encoding

parts of the cDNA translated into protein were considered in this

illustration Thus, exons numbered in this scheme with 1 do not

necessarilycorrespond to exon 1 of the respective gene in the database.

Exons encoding parts of the TRAF domain were summarized and

labeled TD The number of nucleotides encoded byeach exon was

indicated in the box representing the respective exon Exons containing

multiples of three nucleotides are indicated bygrayboxes (B) The

indicated cells were fractionated and lysates containing cytoplasmic

(C) and nuclear proteins (N) were analyzed by Western blotting.

TRAF4 was detected using an affinity-purified polyclonal

anti-(TRAF4) goat Ig recognizing a carboxy-terminal peptide of TRAF4.

Cem T-cells were activated for 6 h with a mixture of PMA (100 n M )

and Ionomycin (1 l ) or remained untreated.

Fig 4 Gel filtration analysis of TRAF4 variants HEK293 cells (20 · 10 6 ) were electroporated with expression plasmids encoding the indicated proteins Two days after transfection, cell lysates (200 lL) were prepared and separated bysize exclusion chromatographyon a HR10/30 Superdex 200 column Fractions of 0.5 mL were collected and analyzed by immunoblotting with a mixture of two GFP/YFP-specifc mAbs Elution volumes of molecular mass standards are indi-cated above.

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the microscope stage To prevent the synthesis of new

protein during the bleaching experiments cycloheximide

(25 lgÆmL)1) was added Fluorescent specimens were

analyzed with a Leica SP2 confocal microscope and imaged

using the LeicaTCSsoftware

R E S U L T S A N D D I S C U S S I O N

Subcellular localization of GFP/YFP-tagged TRAF4

deletion mutants

Using an antiserum against a peptide derived from the

C-TRAF domain of TRAF4, Regnier et al [14] observed

TRAF4 in the nucleus of malignant epithelial cells from

invasive breast carcinomas However, another group

uti-lizing an antiserum generated against a peptide

correspond-ing to the N-terminal 18 amino acids of TRAF4 localized

TRAF4 in the cy toplasm of cells and even failed to detect it

in most breast cancer samples [7] It is possible that these

conflicting results are caused bythe existence of alternative

forms of TRAF4 that could be generated byalternative

splicing or byproteolytic events An alternative possibility

would be that a TRAF4-interacting protein secondarily

regulates TRAF4 localization, eventuallyin a

signal-regu-lated manner To studythe possibilitythat TRAF4 variants

compartmentalize differently, we analyzed the subcellular

localization of GFP or YFP chimeras of TRAF4 deletion

mutants with changed domain architecture (Fig 1) To

determine the cellular localization of the GFP or YFP

fusion proteins, HeLa cells were transientlytransfected with

the respective expression plasmids and analyzed by confocal

microscopythe next day Full-length GFP-tagged TRAF4

molecules were localized in the cytoplasm and were barely

detectable in the nucleus (Fig 2) In a portion of cells

expressing rather high amounts of TRAF4–GFP, the

molecules also accumulated in a few, but large round

patches (data not shown) Such TRAF4–GFP patches are

most likelyto be artifacts caused byoverexpression and are

also found with other TRAF proteins containing a RING–

zinc finger domain (data not shown and Fig 6)

Interest-ingly, both a C-terminal deletion mutant lacking the TRAF

domain [T4(1–268)–YFP] as well as an N-terminal deletion

mutant domain lacking the RING and zinc finger domains

of the molecule [T4(259–470)–YFP]

predominantlylocali-zed to the nucleus (Fig 2) Bysequence analysis Regnier

et al [14] have identified two potential nuclear localization

sequences in the N-terminal part of TRAF4 which are

present in T4(1–268)–YFP However, in T4(259–470)–YFP

there is no obvious nuclear localization sequence TRAF4

deletion mutants lacking the RING domain [T4(75–470)–

YFP] or the C-TRAF domain [T4(1–307)–YFP] showed

also predominant localization in the cytoplasm (Fig 2)

suggesting that the central parts of the molecule (zinc fingers

plus N-TRAF domain) are sufficient to establish

cytoplas-mic retention The nuclear localization of the

aforemen-tioned TRAF4 deletion mutants do not simplyreflect the

deletion of a putative nuclear export sequence, as treatment

for up to 6 h with leptomy cin B, a specific inhibitor of

nuclear export, showed no effect on TRAF4 localization

(data not shown) Thus, the RING and/or C-TRAF

domain seem to be necessaryto localize TRAF4 in the

nucleus Whether this relies on functional nuclear

localiza-tion sequences within these parts of the molecule or on the

interaction with associated proteins remains to be estab-lished Remarkably, analyses of the human TRAF2– TRAF6 genes revealed that all these genes share a stretch

of 3–6 consecutive exons with a multiple of three nucleotides encoding the zinc finger domains of these molecules (Fig 3A) Thus, anycombination of these exons results potentiallyin an in-frame splice form of the respective

Fig 5 Subcellular localization of TRAF4 deletion mutants in cells with cell–cell contacts HeLa cells were seeded on glass cover slides and transientlytransfected with expression constructs for the indicated proteins Next day, representative transfected cells were selected for photography(A) For quantification cells showing increased localiza-tion of the proteins in cell–cell contacts were counted (B).

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TRAF protein In the case of TRAF3, the existence of such

splice forms has indeed alreadybeen described [17,18] It

will be interesting to see in the future whether related splice

forms also exist for TRAF4 and if so, whether these splice

forms exert differential subcellular localization To

investi-gate the subcellular distribution of TRAF4 further we

analyzed cytoplasmic and nuclear extracts with respect to

the content of endogenous TRAF4 Remarkably, in

vari-ance with the overexpression studies discussed above, we

observed in all cell lines investigated (A431, MCF-7, Jurkat,

Cem) that full-length TRAF4 is found mainlyin nuclear

lysates (Fig 3B) In the T-cell lines Jurkat and Cem,

TRAF4 was induced byPMA treatment (see below), but

there were no qualitative differences in these cells in the

distribution of TRAF4 between the cytoplasmic and the

nuclear fraction (data not shown) This discrepancycannot

be attributed to the GFP part of the various TRAF4 fusion

proteins, as several control experiments with nontagged

proteins showed that the GFP part has no influence on the

subcellular distribution of the respective fusion protein (data

not shown) Thus, it is tempting to speculate that a limited

endogenous factor, which is rapidlytitrated

byoverex-pressed TRAF4 forms, is responsible for the putative

nuclear localization of endogenous TRAF4 As shown in

Fig 2, TRAF4 can also be recruited to cell–cell contacts

Thus, it cannot be ruled out that TRAF4 detected in the

nuclear lysates was released from insoluble TRAF4

con-taining cell–cell contact or cytoskeleton structures that

copurifyduring the preparation of the nuclei The Western

blot analyses also regularly revealed a smaller than expected

anti-TRAF4 reactive band, which could represent a splice

form of TRAF4 Additional studies with independent TRAF4 sera should reveal in the future whether this is indeed the case

In gel filtration experiments full-length TRAF4 [T4(1– 470)–GFP], as well as deletion mutants of TRAF4 lacking the Ring domain [T4(75–470)–YFP] or the C-TRAF domain [T4(1–307)–YFP] eluted mainlyin high molecular weight complexes of 443 kDa and more YFP chimeras solelycomprising the N-TRAF domain of TRAF4 [T4(259–307)–YFP] or the complete TRAF domain of the molecule [T4(259–470)–YFP] showed significant complex formation (Fig 4) While the TRAF domain of TRAF4 was almost completelyorganized in complexes, the N-TRAF domain of TRAF4 eluted over the whole fractionation range of the Superdex 200 column A deletion mutant onlycomprising the Ring and zinc finger domain of TRAF4 [T4(1–268)–YFP] eluted over the whole separation range of the gel filtration column, too (Fig 4) The C-TRAF domain of TRAF4 [T4(304–470)–YFP] eluted

as a monomer but has a stabilizing effect on the N-TRAF domain based aggregation of the TRAF domain (Fig 4) A deletion mutant comprising the Ring and zinc finger domain and in addition the N-TRAF domain [T4(1–307)– YFP] eluted predominantlyin high molecular weight fractions Together, these gel filtration data suggest that both the N-TRAF domain and the zinc finger region of TRAF4 drive the formation of TRAF4-containing high molecular weight complexes This is in good accordance with the crystal structures of the TRAF domains of TRAF2 and TRAF3 showing a trimeric trefoil-like structure of these molecules that is mainlybased on the triple helical

Fig 6 Subcellular localization of TRAF1, TRAF2, TRAF3and deletion mutants derived thereof HeLa cells were seeded on glass cover slides and transientlytransfected with expres-sion constructs for the indicated TRAF proteins Representative cells were selected for photography16–36 h after transfection.

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organization of parallel N-TRAF domains [3–5] However,

in principle, it cannot be ruled out completelythat the

separation behavior of the various mutants was caused by

interaction with endogenous TRAF4 or unknown

endo-genous TRAF4-interacting proteins Interaction with

endogenous TRAF4 is most likelynegligible because the

expression level of endogenous TRAF4 in the HEK293 cells

is significantlybelow the expression level of the transfected

constructs (data not shown) but the possible impact of

an endogenous TRAF4-binding protein remains to be

established

As discussed before in isolated cells transfected with T4–

GFP a homogenous cytoplasmic staining was observed In

contrast, in transfected cells that have cell–cell contacts, a

significant local increase of T4–GFP was observed in the

contact sites Analysis of the various TRAF4 deletion

mutants showed that the C-TRAF domain part of the

TRAF domain is sufficient to direct the molecule into the

sites of cell–cell contacts (Fig 5)

To verifywhether other TRAF proteins have a latent

capabilityto translocate to the nucleus similar to

TRAF4, we analyzed the subcellular localization of

GFP-tagged fusion proteins of full-length TRAF1–

TRAF3, and C-terminal as well as N-terminal deletion

mutants derived thereof Similar to TRAF4–GFP, all

other investigated TRAF proteins (T1–GFP, T2–GFP

and T3–GFP) were localized mainlyto the cytoplasm

and were hardlydetectable in the nucleus (Fig 6, left

panel) In contrast to T4(1–268)–YFP, the deletion

mutants of TRAF1–TRAF3 lacking the TRAF domain

still localized in the cytoplasm (Fig 6, middle panel) The

GFP-tagged TRAF domains of TRAF2 and TRAF3

also localized to the cytoplasm whereas the TRAF

domain of TRAF1 showed nuclear and cytoplasmic

localization (Fig 6, right panel) Like all the other

TRAF domains the TRAF domain of TRAF1 is part

of high molecular complex (data not shown) Therefore,

the nuclear localization found for the respective TRAF1

deletion mutant should not be caused bya passive effect

As alreadydiscussed above, round patches with increased

TRAF–GFP concentrations were observable in cells

expressing high amounts of TRAF2– or TRAF3–GFP

(Fig 6) These structures were not found for deletion

mutants solelycomprising the TRAF domain of TRAF2

and TRAF3 but were detected regularlyin cells

trans-fected with TRAF2/3 deletion mutants consisting of the

RING–zinc finger domain

Since TRAF proteins tend to form homo- and/or

heteromers [1,2] we analyzed whether T4–GFP or T4(1–

268)–YFP change their localization upon coexpression with

other nontagged TRAF4 proteins or heterologous TRAF

proteins As shown in Fig 7, coexpression of the nontagged

TRAF domain of TRAF4 [T4(259–470)] was sufficient to

recruit full-length TRAF4 [T4(1–470)–GFP] or T4(75–

470)–YFP into the nucleus whereas cotransfected

non-tagged TRAF4 showed no effect on T4(259–470)–YFP

These data suggest that one T4(259–470) molecule might be

sufficient to direct a heteromeric complex of full-length

TRAF4 and T4(259–470)–GFP into the nucleus TRAF4

deletion mutants lacking the C-TRAF domain [T4(1–307)–

YFP] or onlyconsisting of the N-TRAF domain and the

central part of the C-TRAF domain [T4(259–387)–YFP]

were not recruited into the nucleus upon cotransfection with

T4(259–470) (Fig 7) As both TRAF mutants are able to form high molecular weight complexes, it seems that the N-TRAF domain together with the central part of the C-TRAF domain is sufficient to allow formation of TRAF4-containing complexes but is insufficient to enable nuclear retention byT4(259–470) However, it is unclear whether the deleted C-terminal part of the C-TRAF domain

of T4(259–387)–YFP and T4(1–307)–YFP is necessaryto interact with the TRAF domain of TRAF4 to allow formation of heteromeric complexes or whether the incom-plete C-TRAF domain(s) of these TRAF4 variants lead to

a reduced affinity–avidity of respective heteromeric com-plexes [T4(259–387)–YFP–T4(259–470), T4(1–307) –YFP–

Fig 7 Translocation of TRAF4 from the cytoplasm to the nucleus by T4(259–470) HeLa cells were transfected with the indicated combi-nation of vectors encoding TRAF4, T4–GFP, T4(259–470), T4(259– 470)–YFP, T1–GFP, and GFP–T1(185–416) Next day, representative transfected cells were selected for photography.

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T4(259–470)] for a putative nuclear target structure In

addition, we found no evidence for a

T4(259–470)-depend-ent recruitmT4(259–470)-depend-ent of heterologous TRAFs to the nucleus (data

not shown) In contrast to the TRAF domain of TRAF4,

the TRAF domain of TRAF1 was not able to recruit its

full-length counterpart to the nucleus (data not shown)

Although there was a dominant localization of T4(259–

470)–YFP in the nucleus, a significant part remained in the

cytoplasm (Fig 8) T4(1–470)–GFP was predominantly

found in the cytoplasm but a minor part was detectable in

the nucleus (Fig 8) To verifywhether TRAF4 or the

TRAF4-derived TRAF domain shuttles between nucleus

and cytoplasm, we analyzed T4(1–470)–GFP and T4(259–

470)–YFP byfluorescence loss in photobleaching (FLIP)

Repetitive bleaching for 5–10 times of a small area in the

nucleus depleted the nuclear fluorescence of T4(1–470)–

GFP and T4(259–470)–YFP but had onlya minor effect on

the respective cytoplasmic-located protein fraction (Fig 8)

Correspondingly, fluorescence of cytoplasmic T4(259–470)–

YFP and full-length T4–GFP was alreadysignificantly

reduced after 2 min of bleaching in a small area of the

cytoplasm, whereas the fluorescence of nuclear localized

TRAF4 proteins was almost not affected even after

prolonged bleaching cycles (Fig 8) Together, these data

indicate that there is onlya slow exchange of cytoplasmic

and nucleus-localized TRAF4, indicating that nuclear and

cytoplasmic TRAF4 mayrepresent functionallydistinct populations of this molecule

Analyses of the various deletion mutants of TRAF4 suggest that the zinc fingers of the molecule are responsible for the cytoplasmic retention of TRAF4 Interestingly, it has been recentlyshown that the oncogenic serine–threonine kinase Pim-1 induces translocation of the TRAF4-interact-ing protein-sortTRAF4-interact-ing Nexin 6–TRAF4–associated factor 2 from the cytoplasm to the nucleus [19] Thus, it will be interesting to see in the future whether Pim-1 and sorting Nexin 6 regulate the subcellular distribution of TRAF4 TRAF4 is up-regulated in activated T-cells

In former studies we and others have identified TRAF1 and TRAF2 as possible targets of the NF-jB pathway[20,21]

To find out whether TRAF4 can also be regulated bythis pathway, we treated a variety of cell lines with the potent NF-jB inducers TNF and phorbol 12-myristate 13-acetate (PMA) In most of the investigated cell lines there was no induction, or onlya modest, poorlyreproducible induction,

of TRAF4 mRNA bythese stimuli However, in the T-cell lines Jurkat (Fig 9A) and D23II-7 (Fig 9B) both TNF and PMA/Ionomycin, induced a significant and reproducible up-regulation of TRAF4 mRNA TNF- and PMA-induced TRAF4 expression was alreadydetectable 1 h after

Fig 8 Fluorescence loss in photobleaching (FLIP) analysis of T4(1–470)–GFP and T4(259–470)–YFP HeLa cells were seeded on glass bottom dishes and were transientlytransfected with expression constructs encoding T4(1–470)–GFP and T4(259–470)–YFP Cells were maintained in a conditioned chamber (37 C; 5% CO 2 ) on the microscope stage and areas with two representative cells were selected for FLIP analysis 16–36 h after transfection In one cell GFP fluorescence in the indicated area (white box) of the cell was bleached repetitivelyfor the indicated time (bleaching cell

B) (A) Finally, the average fluorescence intensity (red surrounded areas) in the nucleus (circles) and cytoplasm (boxes) of bleached (filled symbols) and nonbleached (open symbols) (reference cell R) cell was determined using the Leica TCS software (B) and plot against the bleaching time Both, GFP and YFP fusion proteins, were bleached and monitored using 488 nm.

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stimulation in both cell lines, reached its maximum after 3–

6 h and dropped down near to basal levels after 24 h

PMA-induced up-regulation of TRAF4 was also found to a

comparable extent in primaryT-cells and in T-cell blasts

(Fig 9C) Six hours after stimulation, primaryT-cells

showed a 7.8-fold, and day13 T-cell blasts a 6.1-fold,

induction of TRAF4 mRNA Basal TRAF4 mRNA

expression was roughlycomparable in primaryT-cells and

T-cell blasts To verifywhether TRAF4 mRNA is directly

up-regulated byTNF- and PMA-induced signaling

path-ways, we analyzed the effect of the protein synthesis

inhibitor cycloheximide (CHX) on TRAF4 induction We

found no evidence for an inhibitoryeffect of CHX on

TRAF4 up-regulation Moreover, in the presence of CHX

the induction of TRAF4, and also the induction of the

known NF-jB targets TRAF1 and cIAP2, was enhanced

significantly(Fig 9A,B) whereas CHX alone did not

change basal mRNA levels (data not shown) For example,

in the presence of CHX, PMA induced a 15-fold increase of

TRAF4 mRNA in Jurkat cells after 6 h compared to a 2.7-fold induction in the absence of CHX (Fig 9A) Thus, TNF- and PMA-initiated signaling events directlylead to the induction of TRAF4 This is also in good agreement with the rapid kinetics of TRAF4 induction (Fig 9) The increased induction of NF-jB regulated genes in the presence of CHX might reflect that some NF-jB target genes (e.g A20, I-jBa) are involved in the termination of the NF-jB response itself, but this possibilitywas not investi-gated further here

TNF and PMA up-regulate TRAF4 under essential involvement of signaling components of the NF-jB pathway

TNF and PMA/I were chosen for the studies described above, as both are potent inducers of NF-jB To finally verifywhether this pathwayis involved in TRAF4 induction in T-cells, we analyzed a mutant Jurkat cell

Fig 9 TNF and PMA up-regulate TRAF4 mRNA in T-cells Jurkat (A) and D23II-7 T-cells (B) as well as primaryhuman T-cells and human T-cell blasts (C) were stimulated for the indicated times with TNF (20 ngÆmL)1) or a mixture of PMA (100 n M ) and Ionomycin (1 l M ) Jurkat and

D23II-7 cells were analyzed in addition in the presence of CHX (50 lgÆmL)1) that was added 1 h prior to PMA/TNF stimulation Total RNAs were isolated for RPA analysis and 10 lg of each RNA sample were analyzed with a Multi-Probe template set to detect the indicated mRNAs in particular TRAF4 L32 and GAPDH were included as internal controls Protected transcripts were resolved byelectrophoresis on a denaturing polyacrylamide gel (5%) and quantified on a PhosphorImager with the IMAGEQUANT software For quantification each TRAF4 or L32 band was individuallycorrected for background intensities byan area of corresponding size in close neighborhood of the respective mRNA signal To obtain relative TRAF4 and L32 expression values the ratio between the signal intensities of bands of treated cells and the corresponding band of the untreated group were calculated Finally, relative TRAF4 expression values were normalized according to the respective values of relative L32 expresssion The position of protected TRAF4-specific mRNA bands are indicated with an arrow.

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line deficient in expression of IKKc/NEMO [15], an

essential component of the NF-jB-inducing IKK complex

[22] Both TNF and PMA/I-induced up-regulation of

TRAF4 was completelyinhibited in this mutant Jurkat

cell line (Fig 10) Moreover, in a Jurkat clone deficient

for RIP, a molecule involved in TNF but not in

PMA-induced NF-jB activation [16], TNF-PMA-induced but not

PMA-induced TRAF4 expression was blocked (Fig 10)

These data clearlyargue for an essential role of the

NF-jB pathwayin TNF- and PMA-induced up-regulation of

TRAF4

A C K N O W L E D G E M E N T S

We thank Brian Seed (Massachusetts General Hospital, USA) for the

RIP-deficient Jurkat clone and S.-C Sun (Pennsylvania State

Univer-sity, USA) for the IKKc-deficient Jurkat cell line This work was

supported byDeutsche Forschungsgemeinschaft Grant Wa 1025/3–1

and Sonderforschungsbereich 495 project A5.

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H & Iguchi-Ariga, S.M (2001) Pim-1 translocates sorting nexin 6/ TRAF4-associated factor 2 from cytoplasm to nucleus FEBS Lett 506, 33–38.

Fig 10 The NF-jB pathway is involved in TNF- and PMA-induced

up-regulation of TRAF4 Parental Jurkat cells (left panel) or clones derived

thereof deficient in IKKc (middle panel) or RIP (right panel)

expres-sion were stimulated with TNF (20 ngÆmL)1) or PMA (100 n M ) for 6 h

and analyzed by RPA analysis as described in Fig 9.

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