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CHIP did not influence the ATPase cycle of Hsp90 in the absence of co-chaperones or in the pres-ence of the Hsp90 cochaperones Aha1 or p23.. D Single-turnover ATPase rates of Hsc70 and Hs

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preferentially ubiquitinating Hsp70-bound substrates

Marta Stankiewicz1, Rainer Nikolay1,*, Vladimir Rybin2and Matthias P Mayer1

1 Zentrum fu¨r Molekulare Biologie der Universita¨t Heidelberg (ZMBH), DKFZ–ZMBH Alliance, Heidelberg, Germany

2 European Molecular Biology Laboratory, Heidelberg, Germany

Introduction

CHIP consists of an N-terminal TPR (tetratricopeptide

repeat) domain, which binds to the C-terminal EEVD

motif that is present in all cytosolic Hsp70 and Hsp90 chaperones, a central helical domain, which is essential

Keywords

chaperones; Hsp70; Hsp90; protein triage;

ubiquitination

Correspondence

M P Mayer, Zentrum fu¨r Molekulare

Biologie der Universita¨t Heidelberg (ZMBH),

DKFZ–ZMBH Alliance, Im Neuenheimer Feld

282, 69120 Heidelberg, Germany

Fax: +49 6221 545894

Tel: +49 6221 546829

E-mail: M.Mayer@zmbh.uni-heidelberg.de

*Present address

Biochemisches Institut der Universita¨t

Zu¨rich, Winterthurerstrasse 190, 8057

Zu¨rich, Switzerland

(Received 3 May 2010, revised 8 June

2010, accepted 14 June 2010)

doi:10.1111/j.1742-4658.2010.07737.x

The E3 ubiquitin ligase CHIP (C-terminus of Hsc70-interacting protein) is believed to be a central player in the cellular triage decision, as it links the molecular chaperones Hsp70⁄ Hsc70 and Hsp90 to the ubiquitin proteaso-mal degradation pathway To better understand the decision process, we determined the affinity of CHIP for Hsp70 and Hsp90 using isothermal titration calorimetry We analyzed the influence of CHIP on the ATPase cycles of both chaperones in the presence of co-chaperones and a substrate, and determined the ubiquitination efficacy of CHIP in the presence of the chaperones We found that CHIP has a sixfold higher affinity for Hsp90 compared with Hsc70 CHIP had no influence on ADP dissociation or ATP association, but reduced the Hsp70 cochaperone Hdj1-stimulated sin-gle-turnover ATPase rates of Hsc70 and Hsp70 CHIP did not influence the ATPase cycle of Hsp90 in the absence of co-chaperones or in the pres-ence of the Hsp90 cochaperones Aha1 or p23 Polyubiquitination of heat-denatured luciferase and the native substrate p53 was much more efficient

in the presence of Hsc70 and Hdj1 than in the presence of Hsp90, indicat-ing that CHIP preferentially ubiquitinates Hsp70-bound substrates

Structured digital abstract

l MINT-7904367 : CHIP (uniprotkb: Q9UNE7 ) and HSP 90-beta (uniprotkb: P08238 ) physically interact ( MI:0915 ) by molecular sieving ( MI:0071 )

l MINT-7904785 : HSP 90-beta (uniprotkb: P08238 ) and p23 (uniprotkb: Q15185 ) bind ( MI:0407 ) by molecular sieving ( MI:0071 )

l MINT-7904047 : CHIP (uniprotkb: Q9UNE7 ), HSP 90-beta (uniprotkb: P08238 ) and p23 (uni-protkb: Q15185 ) physically interact ( MI:0915 ) by molecular sieving ( MI:0071 )

l MINT-7903424 : Alpha-lactalbumin (uniprotkb: P00711 ), HSP70 (uniprotkb: P08107 ) and CHIP (uniprotkb: Q9UNE7 ) physically interact ( MI:0915 ) by molecular sieving ( MI:0071 )

l MINT-7903354 : CHIP (uniprotkb: Q9UNE7 ) and HSC70 (uniprotkb: P11142 ) bind ( MI:0407 )

by isothermal titration calorimetry ( MI:0065 )

l MINT-7903373 : CHIP (uniprotkb: Q9UNE7 ) and HSP90-beta (uniprotkb: P08238 ) bind ( MI:0407 ) by isothermal titration calorimetry ( MI:0065 )

Abbreviations

CHIP, C-terminus of Hsc70-interacting protein; Hsp70, 70 kDa heat shock protein; Hsp90, 90 kDa heat shock protein; Hsc70, 70 kDa heat shock cognate; ITC, isothermal titration calorimetry; MABA-ADP ⁄ MABA-ATP, N 8 -(4-N¢-methylanthraniloylaminobutyl)-8-aminoadenosine 5¢-di ⁄ triphosphate; RCMLA, reduced carboxymethylated a-lactalbumin; TPR, tetratricopeptide repeat.

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for dimerization, and a C-terminal U-box domain,

which is responsible for interaction with E2

ubiquitin-conjugating enzymes [1,2] This interaction with

molec-ular chaperones suggests that CHIP is involved in the

triage decision [3–5] Hsp70 chaperones are essential

components of the cellular quality control network,

interacting with virtually all misfolded proteins,

pre-venting their aggregation and assisting their refolding

into the native state [6–8] Hsp90 chaperones have also

been shown to bind to misfolded proteins [9], but their

main essential function in all eukaryotic cells is

believed to be interaction with a large number of

regu-latory proteins, called client proteins, including

recep-tors, protein kinases and transcription factors [10–12]

The question therefore arises as to whether CHIP

pref-erentially interacts with Hsp70 to mark misfolded

proteins, which cannot be refolded to the native

state, for degradation, or whether it assumes a more

regulatory role by interacting mainly with Hsp90 to

ubiquitinate signaling proteins

It has been shown in vitro and in cell culture that

sev-eral Hsp90 clients are ubiquitinated in a

CHIP-depen-dent manner, and that this ubiquitination depends on

CHIP’s U-box domain [13–17] In some cases, CHIP

appears to directly bind its substrates and ubiquitinate

them in a chaperone-independent manner [18–20] Such

specific substrate recognition is a typical feature of E3

ubiquitin ligases; however, the majority of CHIP’s

sub-strates described so far are bona fide Hsp70 and Hsp90

clients, and their degradation depends on the presence

of the chaperones [13,21–23] Among the CHIP

substrates are many regulatory proteins that are

ubiqui-tinated and degraded even in the presence of an Hsp90

inhibitor such as geldanamycin [21]

For most if not all functions, Hsp90 cooperates with

Hsp70 in a chaperone cycle, which was first proposed

for steroid hormone receptors and involves a number

of co-chaperones [24,25] Steroid hormone receptors

first interact with Hsp40 and Hsp70 The dimeric

pro-tein Hop (Hsp70-Hsp90 organizing Propro-tein), which has

separate TPR domains for binding to Hsp70 (TPR1)

and Hsp90 (TPR2a), assembles the early client

com-plex with Hsp70 and Hsp90 Hop and Hsp70 are then

replaced by p23 and a TPR domain-containing

pept-idyl-prolyl-cis⁄ trans-isomerase (e.g the 51 and 52 kDa

FK506-binding proteins FKBP51 or FKBP52) The

mature complex decays with a half life of

approxi-mately 5 min, and the hormone receptor re-enters the

cycle by binding to Hsp40 and Hsp70 As CHIP can

interact with Hsp70 and Hsp90, it is not clear whether

ubiquitination of the chaperone substrate occurs while

the substrate is bound to Hsp70 or Hsp90 Another

intriguing question is how the triage decision is made

As CHIP is a TPR-containing co-chaperone, it com-petes with numerous other TPR-containing co-chaper-ones for binding to Hsp70 and Hsp90 [26]

Here we provide new insights into the triage decision

by assessing the physical interaction of CHIP with Hsp70⁄ Hsc70 and Hsp90, and analyzing the functional consequences for the chaperone substrates

Results CHIP–chaperone interaction and competition with Hop

To determine how the triage decision is made, we first addressed the question of protein affinities and cellular concentrations CHIP directs proteins to the degrada-tion pathway, and Hop is an essential co-chaperone for protein folding Because they both interact with the same C-terminal EEVD motif of Hsc70 and Hsp90, we determined the affinities of CHIP for Hsc70 and Hsp90 using isothermal titration calorimetry (ITC) (Fig 1A,B) We also investigated the interaction

of CHIP with heat shock-induced Hsp70, to determine whether CHIP has a preference for this homolog to enhance quality control processes during heat shock CHIP’s affinity for Hsp90 (KD= 0.38 ± 0.04 lm) was approximately six times higher than that for Hsc70 (KD= 2.3 ± 0.3 lm), and two and a half times higher than that for Hsp70 (KD= 0.95 ± 0.01 lm) The affinities of CHIP for Hsc70 and Hsp70 were in the same range as the value measured for the Hop– Hsc70 interaction (KD= 1.5 ± 0.2 lm) using surface plasmon resonance spectroscopy [27] In contrast, for the interaction of Hop with Hsp90, a KD value

of 0.1 ± 0.02 lm was determined by surface plasmon resonance spectroscopy, which is only one quarter of the value measured here for the CHIP–Hsp90 interac-tion These results indicate that Hop and CHIP com-pete efficiently with each other for binding to Hsc70⁄ Hsp70 when the C-termini of Hsc70 ⁄ Hsp70 are limiting when the concentration of Hsc70/Hsp70 is lower than the combined concentration of CHIP and Hop, but Hop appears to be at an advantage com-pared to CHIP when binding to Hsp90

The cellular concentrations of Hsc70⁄ Hsp70, Hsp90, CHIP and Hop where determined by quantitative Wes-tern blot using HEK293, a commonly used epithelial cell line, and Jurkat cells, which are a model for acute T-cell leukemia (Fig 1C and Table 1) The values determined for Hsc70⁄ Hsp70 (0.9 and 0.4% of total protein for HEK293 and Jurkat cells, respectively) and Hsp90 (0.6 and 0.8% of total protein for HEK293 and Jurkat cells, respectively) were somewhat lower than

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the values of 1–2% reported for other cell lines [27a].

For Hop, we determined a relative amount of 0.2% in

both cell lines In contrast, the relative amount of

CHIP varied significantly, being 0.07% in HEK293

and only 0.01% in Jurkat cells As the total protein

concentration was 154 mgÆmL)1 in HEK293 cells [28]

and 127 mgÆmL)1 in Jurkat cells [29], the

concentra-tions of Hsc70⁄ Hsp70 are 20 and 7 lm, those of

Hsp90 are 11 and 12 lm, those of Hop are 5 and

4 lm, and those of CHIP are 3.1 and 0.4 lm in

HEK293 and Jurkat cells, respectively Given these

concentrations of Hsc70, Hsp90, CHIP and Hop and

the KD values, approximately 8.5 and 1.4% of Hsc70

and 4.6 and 0.7% of Hsp90 molecules have CHIP

bound to their C-termini at any given moment in

HEK293 and Jurkat cells, respectively As there are a large number of TPR domain proteins in higher eukaryotic cells, many of which bind to Hsp90 with a similar affinity as CHIP and Hop do [30,31], the amount of Hsp90 occupied by CHIP is probably much lower than estimated above Fewer TPR proteins have been shown to bind Hsc70⁄ Hsp70 [26] Therefore, based on our affinity determination and quantitative Western blots, the amount of Hsc70 occupied by CHIP is estimated to be 1–9% (in Jurkat and HEK293 cells)

Influence of CHIP on substrate binding of Hsp70 RING and U-box E3 ligases do not transfer ubiquitin themselves but generally bring substrates and E2 ubiquitin-conjugating enzymes in close proximity by binding to both proteins It has been shown that CHIP has the ability to bind substrates [18–20] If CHIP also contacts substrates when bound to Hsp70, it might increase the stability of the Hsp70–substrate complex, thereby allowing more time for ubiquitin transfer by the E2 enzyme

We therefore assessed whether CHIP affects the equilibrium dissociation constant (KD) or the dissocia-tion rate constant (koff) of the Hsp70–substrate complex by analyzing the formation of complexes of Hsp70 with reduced carboxymethylated a-lactalbumin

Fig 1 Interaction of CHIP with Hsc70 and

Hsp90 and in vivo concentrations of the

chaperones and co-chaperones (A,B)

Deter-mination of the interaction parameters of

the CHIP–Hsc70 (A) and CHIP–Hsp90 (B)

complexes using isothermal titration

calorim-etry (C) Quantitative immunoblot for

deter-mination of the in vivo concentrations of

Hsp70 ⁄ Hsc70, Hsp90, CHIP and Hop in

HEK293 and Jurkat cells Various amounts

of purified protein (15–400 ng, left panels)

and cleared protein extracts (10–100 lg) of

HEK293 (middle panels) and Jurkat cells

(right panels), as indicated, were separated

by SDS ⁄ PAGE and analyzed by

immunoblot-ting with specific antisera The upper bands

detected in vivo for CHIP and Hop most

likely represent phosphorylated variants of

the proteins [72,73].

Table 1 Relative amounts of CHIP, Hop, Hsp70 and Hsp90 in

HEK and Jurkat cells The relative amounts of chaperone and

co-chaperones were determined by quantitative immunoblotting as

shown in Fig 1C using purified proteins as standards.

Percentage of

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(RCMLA), a model chaperone substrate [32], in the

presence and absence of CHIP using gel filtration and

3H-RCMLA (Fig 2A,B) When Hsp70 was

pre-incu-bated with CHIP, the amount of RCMLA bound to

Hsp70 decreased by approximately 40% (Fig 2B)

This was not observed with the CHIP-K30A variant,

which does not bind to the C-terminal EEVD motif of

Hsp70 and Hsp90 [21], suggesting that CHIP does not

compete for Hsp70’s substrate binding pocket but

affects the chaperone–substrate interaction in an

indi-rect way This result clearly indicates that CHIP does

not prolong the half life of the high-affinity Hsp70–

chaperone complex

To investigate substrate release, we chased the

pre-formed Hsp70–3H-RCMLA complexes with unlabeled

RCMLA in the absence and presence of CHIP As

shown in Fig 2C, CHIP did not influence substrate

release by Hsp70, suggesting that the decrease in

detectable Hsp70–RCMLA complex is due to a

decreased association rate CHIP bound to the

C-ter-minus of Hsp70 possibly creates a steric hindrance to

substrate binding We did not observe any direct

inter-action of CHIP with the chaperone substrate RCMLA,

suggesting that substrate binding by CHIP may be a

specific interaction limited to certain proteins In

con-clusion, CHIP did not increase the half life of the

Hsp70–RCMLA complex by directly stabilizing the

chaperone–substrate interaction, but instead decreased

the amount of Hsp70-bound RCMLA by 40%

Influence of CHIP on the ATPase cycle of Hsp70

As nucleotide exchange by Hsp70 is rate-limiting for

substrate release under physiological ATP

concentra-tions, CHIP could also affect the half-life of the

Hsp70–substrate complex by altering the ATPase cycle

of Hsp70 proteins It has been reported that CHIP

decreases the ATPase rate stimulated by the J-domain

co-chaperones Hdj1 and Hdj2 but not the basal

ATPase rate under steady-state conditions [14,33] In

the absence of a J-domain co-chaperone, c-phosphate

cleavage is rate-limiting in the ATPase cycle of Hsp70

proteins [34,35] In the presence of a J-domain protein,

nucleotide exchange becomes rate-limiting [36,37] The

CHIP-induced reduction of the Hdj1⁄ Hdj2-stimulated

ATPase rate of Hsc70 could therefore be caused by a

reduced nucleotide exchange, which in turn would

increase the dwell time of the substrate on the Hsp70

chaperone To address this point, we analyzed the

influence of CHIP on ADP dissociation from and

ATP association with Hsc70 and Hsp70 using the

fluorescent nucleotide analogs N8

-(4-N¢-methylanthra-niloylaminobutyl)-8-aminoadenosine 5¢-di ⁄ triphosphate

(MABA-ADP⁄ MABA-ATP) [38] and stopped-flow instrumentation To measure the basal ADP dissocia-tion rate, Hsc70 or Hsp70 were pre-incubated with MABA-ADP in the absence or presence of a 20-fold excess of CHIP, and subsequently mixed with an

Fig 2 CHIP reduces the affinity of Hsp70 for a model substrate without affecting the dissociation rate (A) Size-exclusion chroma-tography of 3 H-RCMLA (reduced carboxymethylated a-lactalbumin) after pre-incubation in the absence or presence of Hsp70 and CHIP

as indicated (B) Quantification for the size-exclusion chromatogra-phy experiments shown in (A) 3 H-RCMLA was pre-incubated with the indicated proteins The amount of radioactivity in elution volume 9–11.5 mL, in which the RCMLA–Hsp70 complex elutes, is shown relative to the radioactivity in elution volume 12–15.5 mL, in which free RCMLA elutes (C) Dissociation of the RCMLA–Hsp70 com-plex.3H-RCMLA was pre-incubated with Hsp70 before addition of CHIP where indicated At time point 0, a fivefold excess of unla-beled RCMLA was added The complex was analyzed by size-exclu-sion chromatography at various time points The dissociation rate constants (koff) were determined by fitting a single exponential decay function to the data The inset shows the dissociation rate constants in the absence and presence of CHIP (mean ± SEM of two independent determinations).

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excess of ATP As shown in Fig 3A, CHIP had no

influence on the basal ADP dissociation rates of Hsc70

and Hsp70 These results did not explain the

CHIP-mediated decrease in the Hdj1⁄ Hdj2-stimulated steady-state ATPase rate

In vivo nucleotide exchange factors such as Bag-1 accelerate ADP dissociation by Hsc70 and Hsp70 [37,39] It has been reported that CHIP and Bag-1 interact with each other [40] We therefore determined whether CHIP could influence Bag-1-stimulated nucle-otide exchange To address this question, we pre-incu-bated Hsc70⁄ Hsp70 with MABA-ADP in the absence and presence of a large excess of CHIP, and subse-quently rapidly mixed the reaction mixture with Bag-1 and an excess of ATP As expected, Bag-1 stimulated the ADP dissociation rate by approximately 20-fold at stoichiometic concentrations Even a large excess of CHIP only slightly decreased the Bag-1-stimulated ADP dissociation rate of Hsc70 and Hsp70 (Fig 3A)

No effect of CHIP on the Bag-1-stimulated ADP dis-sociation rate was observed when CHIP was added together with Bag-1 instead of pre-incubated with the Hsp70 protein (data not shown) Therefore, the reported interaction of CHIP and Bag-1 has no strik-ing effect on the nucleotide release function of Bag-1

To analyze the second step of nucleotide exchange, ATP association, we pre-incubated Hsc70 or Hsp70 in the absence and presence of Bag-1 and a 20-fold excess

of CHIP, and subsequently mixed the reaction mixture with MABA-ATP As shown in Fig 3B,C, CHIP did not slow down ATP association significantly in the absence or presence of Bag-1 Instead we observed a slight increase in ATP association rate for Hsc70 in the presence of CHIP

As neither ADP dissociation nor ATP association are negatively affected by CHIP, the reduction in the ATPase activity must be due to an effect on c-phos-phate cleavage To verify this hypothesis, we performed single-turnover ATPase experiments The basal ATPase rate of Hsp70 proteins is very low but can be stimulated

by a J-domain co-chaperone at high concentrations (> 10-fold) As shown in Fig 3D, high concentrations

of CHIP had no effect on the basal single-turnover ATPase rate but decreased the Hdj1-stimulated ATPase

Fig 3 CHIP affects Hdj1-stimulated c-phosphate cleavage by Hsc70 ⁄ Hsp70 but not nucleotide exchange (A) MABA-ADP dissoci-ation rates of Hsc70 and Hsp70 in the absence and presence of CHIP and Bag-1 (B) Fluorescence traces of MABA-ATP association with Hsc70 in the absence and presence of CHIP and Bag-1 (C) Association rates for MABA-ATP in the absence and presence of Bag-1 and CHIP The columns show the rates for the fast phase (k1) and the slow phase (k2) of a fit of a two-phase exponential equation to the traces in (B) and additional data (D) Single-turnover ATPase rates of Hsc70 and Hsp70 in the absence and presence of Hdj1 and CHIP as indicated.

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rate, consistent with previous steady-state ATPase data

[14,33] Taken together, we found no evidence that

CHIP influences the chaperone cycle of Hsp70 proteins

to prolong the life-time of the substrate–Hsp70–CHIP

complex and thereby to increase the possibility of

recruitment of the E2 ubiquitin-conjugating enzyme and

ubiquitination In contrast, CHIP decreased the

Hdj1-triggered c-phosphate cleavage, thereby

decelerat-ing transition from the low-affinity to the high-affinity

substrate-binding state

Influence of CHIP on the ATPase activity and

co-chaperone binding of Hsp90

We next determined whether CHIP influences the

ATPase cycle of Hsp90 in order to increase the

possibil-ity of ubiquitination of an Hsp90-bound client protein

We performed steady-state ATPase assays of Hsp90 in

the absence and presence of CHIP, and in the absence

and presence of Aha1 and p23, two co-chaperones that

are known to influence the ATPase activity of Hsp90

We obtained a value of 1.2 ± 0.1· 10)3s)1 for the

basal ATPase activity of Hsp90 This rate was

stimu-lated fivefold by a threefold excess of Aha1 over Hsp90,

and inhibited to 50% of the basal rate by a tenfold

excess of p23, consistent with published data [41,42] As

shown in Fig 4, CHIP did not significantly affect the

basal ATPase rate of Hsp90, and also had no influence

on the Aha1-stimulated or p23-inhibited rate, even at a

tenfold excess over Hsp90

A previous study suggested that p23 competes with

CHIP for binding to Hsp90 [13] As CHIP did not

reduce the inhibitory effect of p23 on the ATPase

activity of Hsp90, we determined whether this is due

to the inability of CHIP to bind to Hsp90 in the

pres-ence of p23 We therefore incubated Hsp90 with CHIP

and p23 and used gel filtration to analyze the

com-plexes formed As evident from Figs 5A,B and S1,

CHIP forms a stable complex with Hsp90 and p23 and

does not prevent p23 binding to Hsp90 In contrast,

Hop reduced binding of p23 to Hsp90, consistent with

previous observations [43] Aha1 also reduced binding

of p23 to Hsp90, and CHIP could not reverse this

effect of Aha1 Taken together, CHIP did not reduce

the ATP hydrolysis rates of Hsp90 As ATP hydrolysis

leads to substrate release [44], CHIP should not

increase the half-life of Hsp90–client complexes

Unfolded proteins are more efficiently

ubiquitinated in the presence of the Hsp70 system

As CHIP interacts with both Hsp70 and Hsp90 and

the interaction is mutually exclusive, we wished to

directly compare the two chaperone systems in terms

of their influence on the efficiency of CHIP-mediated ubiquitination of a substrate It has already been shown, that both systems are able to support ubiqui-tination in vitro, but quantitative time-resolved ubiq-uitination experiments are necessary to compare their relative efficiency We pre-incubated the chaperone substrate firefly luciferase in the presence of various concentrations of Hsc70 plus Hdj1 or Hsp90 at

43C, and subsequently shifted the temperature to

30C before adding CHIP, the E2 enzyme UbcH5c, the E1 ubiquitin-activating enzyme and ubiquitin In the presence of Hsc70 and Hdj1, ubiquitination was very efficient even at low chaperone:luciferase ratios

Fig 4 CHIP has no influence on the ATPase rate of Hsp90 (A) Steady-state ATPase rate of Hsp90 in the absence or presence of the indicated concentrations of CHIP (B) Steady-state ATPase rate

of Hsp90 in the absence or presence of the indicated concentra-tions of p23 and CHIP (C) Steady-state ATPase rate of Hsp90 in the absence or presence of the indicated concentrations of Aha1 and CHIP.

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(1 : 1), but not in the absence of Hdj1 (Fig 6A,B, left

panel) In contrast, ubiquitination in the presence of

Hsp90 was not very efficient and required high

con-centrations of chaperone This indicates that many

more substrate molecules can be successfully

ubiquiti-nated per one Hsc70⁄ CHIP complex than per one

Hsp90⁄ CHIP complex Time-resolved experiments

also showed that CHIP-mediated polyubiquitination

was faster in the presence of the Hsc70⁄ Hdj1 system than in the presence of Hsp90 (Fig 6B, right panel) Interestingly, when we used the lysine-free variant of ubiquitin (Ubi-K0) to prevent polyubiquitination, we also detected multiple bands of luciferase in the pres-ence of Hsc70 and Hsp90, indicating that both chap-erones allow attachment of ubiquitin to several lysines of luciferase These data suggest that, several lysines of the substrate are modified even in the pres-ence of wild-type ubiquitin As multiple bands of ubiquitinated luciferase are already visible at the

20 min time point in the case of wild-type ubiquitin but appear later in the case of the K0 ubiquitin vari-ant, polyubiquitination may occur in the presence of Hsp70 with a certain processivity, or alternatively the lysines in ubiquitin (presumably Lys48) are better substrates for ubiquitination than lysines in the sub-strate

In addition, the co-chaperones of Hsp70 regulate the reaction in a dynamic manner Hdj1 strongly enhanced ubiquitination, as mentioned above (Fig 6A), but Bag-1 reduced the ubiquitination efficacy (Fig 6C, left panels) In contrast, neither p23 nor Aha1 had an impact on the basal Hsp90-dependent ubiquitination (Fig 6C, right panels) The presence of Hop reduced the ubiquitination of luciferase for both Hsc70 and Hsp90; however, the effect was observed only after shorter time periods, and polyubiquitinated species accumulate after longer time periods, despite increas-ing Hop concentrations (Fig 6D) Both systems gener-ate substrgener-ates with multiple ubiquitingener-ated sites, as shown for the reaction using a lysine-free ubiquitin mutant (Fig 6B) However, unfolded proteins are more efficiently ubiquitinated in the presence of the Hsp70 system

Ubiquitination of a native chaperone substrate protein

Hsp70 and Hsp90 not only interact with misfolded proteins but also with native or near-native proteins

To investigate CHIP-mediated ubiquitination of a native protein substrate, we chose the tumor suppres-sor p53, which has been shown to interact with Hsp70 and Hsp90 [45,46] At 25C, p53 was efficiently mono-ubiquitinated by CHIP in the absence of chaper-ones (Fig 7A) Neither Hsc70⁄ Hdj1 nor Hsp90 enhanced this ubiquitination reaction At 37C, Hsc70⁄ Hdj1 but not Hsp90 stimulated CHIP-mediated polyubiquitination of p53 Aha1 and p23 had only minor effects on CHIP-mediated ubiquitination in the presence of Hsp90 (Fig 7B) Taken together, as in the case of luciferase, ubiquitination of the native

Fig 5 Influence of CHIP on p23 binding to Hsp90 Hsp90 and p23

were incubated in the absence or presence of CHIP, Hop and Aha1

as indicated, and subsequently separated by size-exclusion

chroma-tography on a Superose TM 12 10 ⁄ 300 column (GE Healthcare,

Frei-burg, Germany), and analyzed by SDS ⁄ PAGE and Coomassie Blue

staining (A) Representative SDS gels (B) Quantification of the gels

shown in (A), Fig S 1 and additional data: the bands representing

p23 were quantified in all lanes The bar graph shows the amount

of p23 co-eluting with Hsp90 (lanes 4–6) relative to the total

amount of p23 (sum of all lanes).

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chaperone substrate p53 was more efficient in the

pres-ence of Hsc70 and Hdj1 than in the prespres-ence of Hsp90

and its co-chaperones Aha1 or p23

Discussion

Our study shows that CHIP cooperates with Hsc70

and Hsp90 in a rather passive manner As CHIP has

no effect on substrate dissociation, ADP dissociation

or ATP association, it does not increase the half-life of

an Hsp70–substrate complex to provide more time for recruitment of the E2 ubiquitin-conjugating enzyme Similarly, CHIP had no influence on the ATPase cycle

of Hsp90 to prolong the bound state, the ATP-bound state has a high affinity for substrates We con-clude that CHIP to sample available Hsc70–substrate and Hsp90–substrate complexes in a stochastic process and thereby occasionally effects ubiquitination Sub-strates that are efficiently folded or refolded and there-fore spend a relatively short time in complex with Hsc70 or Hsp90 have only a small chance of being ubiquitinated In contrast, substrates that cannot be folded efficiently and consequently cycle on and off the chaperones continuously, or remain bound to chaper-one for a prolonged time interval, will eventually be ubiquitinated by CHIP and targeted for degradation Such substrates may be misfolded proteins such as heat-denatured luciferase, which we have used in our study, or de novo folding substrates as the cystic

Fig 6 CHIP-mediated ubiquitination of a denatured substrate is more efficient in the presence of Hsc70 and Hdj1 than in the pres-ence of Hsp90 (A–D) Immunoblots of SDS ⁄ PAGE -separated ubiq-uitination reactions using a luciferase-specific antiserum (A) Time course of ubiquitination of heat-denatured firefly luciferase in the presence of Hsc70 and in the presence and absence of Hdj1 Heat-denatured firefly luciferase was ubiquitinated in the presence of

50 n M E1, 1 l M UbcH5c, 1 l M CHIP (except lane 1), 100 l M ubiqu-itin (except lane 2) and 5 l M Hsc70, in the absence (lanes 3–8) and presence (lanes 9–14) of 5 l M Hdj1 for 1–20 min as indicated (B) Comparison of CHIP-dependent poly- and multi-ubiquitination effi-ciency in the presence of Hsc70 ⁄ Hdj1 and Hsp90 Left panel, ubiq-uitination of firefly luciferase at various concentrations of Hsc70 (0.2–6 l M ) with 5 l M Hdj1 and various concentrations of Hsp90 (0.2–6 l M ) as indicated Luciferase was heat-denatured in the pres-ence of the chaperones, and the ubiquitination mix consisting of

50 n M E1, 1 l M UbcH5c, 1 l M CHIP and 100 l M ubiquitin was added Right panel, CHIP-dependent ubiquitination of heat-dena-tured luciferase in the presence of 5 l M Hsc70 plus 5 l M Hdj1 (lanes 9–16) or 5 l M Hsp90 (lanes 17–24) with wild-type ubiquitin (lanes 9–12 and 17–20) or the lysine-free ubiquitin variant Ubi-K0, in which all lysines were replaced by arginines (lanes 13–16 and 21–24) for 10–80 min as indicated (C) Ubiquitination of firefly lucif-erase in the presence of various chaperones and co-chaperones Lanes 1–12: ubiquitination of luciferase in the presence of 5 l M Hsc70 plus 5 l M Hdj1 and the absence (lanes 1–6) or presence (lanes 7–12) of 5 l M Bag-1 for 5–120 min as indicated Lanes 13 to 25: ubiquitination of luciferase in the presence of 5 l M Hsp90 and the absence of co-chaperones (lanes 13–17) or the presence of

5 l M Aha1 (lanes 18–21) or 5 l M p23 (lanes 22–25) for 5–40 min as indicated Lane 13 shows the ubiquitination of luciferase in the absence of CHIP but the presence of Hsp90 (D) CHIP-dependent ubiquitination of heat-denatured firefly luciferase in the presence of

5 l M Hsc70 plus 5 l M Hdj1 (lanes 1–5) or 5 l M Hsp90 (lanes 6–16) and increasing concentrations of Hop (0–23 l M ) for 20 and 120 min

as indicated.

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fibrosis transmembrane regulator CFTR, a

slow-fold-ing variant (CFTRDF508) of which is known to be

efficiently degraded and has been shown to be

ubiquiti-nated in a CHIP-dependent way [14] Such a

mecha-nism is also consistent with the phenotype of

CHIP) ⁄ )-knockout mice, which accumulate aggregated

proteins [47] The small amount of proteins that are

ubiquitinated erroneously is the price to be paid for

efficient quality control Such a mechanism is

reminis-cent of the quality control in the endoplasmic

reticu-lum, where newly synthesized glycoproteins are folded

in the calnexin⁄ calreticulin cycle [48] Misfolded

glyco-proteins are bound in turn by the chaperones calnexin

or calreticulin and the folding sensor

UDP-glucose-glycoprotein-glucosyltransferase Proteins that fold

properly exit this cycle Glycoproteins that remain in the cycle for an extended period of time have a high probability that their N-linked glycans will be trimmed

by a-1,2-mannosidase I, marking the protein for degra-dation by the ER-associated degradegra-dation pathway The affinity of dimeric CHIP for dimeric Hsp90 (0.38 lm) was approximately sixfold higher than its affinity for monomeric Hsc70 (2.3 lm) This result sug-gests binding of the dimeric CHIP to both C-termini

of the dimeric Hsp90, in agreement with a recent amide hydrogen exchange study analyzing the interac-tion of Hsc70 and Hsp90 with CHIP [49] A KD of 2.4 lm was found previously for the interaction of CHIP with a peptide comprising the ten C-terminal residues of Hsp90 [1] This value most likely represents the KDfor the initial binding of one TPR domain to a single EEVD motif of the Hsp90 dimer in a two-step sequential binding mechanism KD values in the high nanomolar range have also ben obtained for the inter-action of other TPR proteins with Hsp90 [27,30] Therefore, TPR domain proteins compete efficiently with CHIP for binding to Hsp90, and only a small amount of Hsp90 is bound to CHIP at equilibrium This contrasts with the situation for Hsc70⁄ Hsp70, whose C-termini interact with only the TPR domain proteins Hop and CHIP [26] As the concentration of Hsc70 is greater than the concentrations of Hop and CHIP together, changes in the CHIP concentration change the concentration of the Hsc70–CHIP complex, making the system very sensitive to CHIP concentra-tions Despite the lower affinity of CHIP for Hsc70 compared to Hsp90, CHIP is more frequently in com-plex with Hsc70 in the cell than with Hsp90

We further demonstrate that ubiquitination of heat-denatured luciferase is much more efficient in the pres-ence of Hsc70 and Hdj1 than in the prespres-ence of Hsp90 This observation suggests that misfolded proteins at least are targeted to the ubiquitin⁄ proteasomal path-way through the Hsp70 system rather than through the Hsp90 system Such a mechanism might also be true for bona fide Hsp90 clients once an Hsp90-specific inhibitor is added This has been indicated by data for the glucocorticoid receptor, which was found to co-localize with Hsp70 and CHIP after addition of gel-danamycin but not with Hsp90 and FKBP52 [50] However, if CHIP is over-expressed ectopically or as a consequence of a pathological process, even Hsp90-bound clients may be ubiquitinated and degraded [13– 17] As all Hsp90 clients, which are degraded upon CHIP over-expression, are also substrates of Hsc70, the overproduced CHIP may act on the Hsc70–client complex rather than the Hsp90–client complex Our data with p53 support this hypothesis Therefore, it

Fig 7 Chip-mediated ubiquitination of the native chaperone

sub-strate p53 (A) Temperature dependence of p53 ubiquitination p53

was ubiquitinated in the absence and presence of Hsc70⁄ Hdj1,

Hsp90 or both at 25 C (left panel) or 37 C (right panel)

Immuno-blot of SDS ⁄ PAGE-separated ubiquitination reactions using a

p53-specific antiserum (B) The Hsp90 co-chaperones Aha1 and

p23 have no influence on CHIP-mediated ubiquitination of p53 p53

was ubiquitinated in the presence of Hsp70 ⁄ Hdj1, Hsp90 and Aha1

and p23 as indicated.

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remains unclear whether CHIP can selectively

ubiquiti-nate folded Hsp90-associated clients, thereby

perform-ing regulatory functions in the cell If such a function

of CHIP exists, it seems to be of minor importance, as

the stability of several bona fide Hsp90 substrates is

not affected in CHIP) ⁄ ) mouse embryonic fibroblasts

[51] The CHIP) ⁄ ) mice show phenotypes related to

abnormal protein aggregation [47] rather than

break-down of signaling pathways (compare with FKBP52

knockout mice [52,53]) All these facts speak in favor

of CHIP being an E3 ubiquitin ligase with low

sub-strate specificity that is responsible for the clearance of

hopeless cases of protein folding However, the role of

CHIP in direct substrate binding and its E4 ligase

function [54,55] remain puzzling

The native chaperone substrate p53 was only

mono-ubiquitinated by CHIP at 25C, and the chaperones

did not enhance this ubiquitination nor stimulate

poly-ubiquitination at this temperature At 37C,

Hsc70⁄ Hdj1 but not Hsp90 stimulated CHIP-mediated

polyubiquitination NMR experiments with p53 core

domain showed that the core domain starts unfolding

at 37C and binds concomitantly to Hsp90 [56] The

Hsc70⁄ Hdj1-stimulated polyubiquitination may

there-fore be due to recognition of unfolded regions within

p53 by the chaperone Therefore, ubiquitination of p53

appears to be very similar to ubiquitination of the

denatured firefly luciferase

Our study also clarified the previously observed

effect of CHIP on the Hdj1⁄ Hdj2-stimulated

steady-state ATPase rate of Hsc70 [14,33] We show here that

nucleotide exchange of Hsc70 is not affected by CHIP

In contrast, CHIP decreased the Hdj1-stimulated

c-phosphate cleavage, as demonstrated by

single-turn-over ATPase experiments CHIP slows down the

transition of Hsc70 from a low-affinity state with high

substrate association and dissociation rates to a

high-affinity state with low substrate dissociation rates

CHIP therefore counteracts the targeting function of

the J-domain protein The molecular basis for this

observation could be a reduced association rate for

substrates It was shown previously that Hsp70

pro-teins require two signals for highly efficient hydrolysis

of ATP: one signal provided by the J-domain and a

second signal provided by the substrate [57–62] High

concentrations of some J-domain proteins can provide

both signals by interaction with the substrate binding

pocket as well as the ATPase domain [59,63–65] As

CHIP reduces the affinity for substrates without

affect-ing the dissociation rate, substrate association is

conse-quently reduced This in turn reduces the substrate

signal for ATP hydrolysis It may be advantageous if

substrates do not associate with Hsc70 when CHIP is

already bound Such a mechanism would prevent ubiq-uitination of a substrate that has not had the opportu-nity to refold

In summary, our results suggest the model shown in Fig 8 Proteins in an intermediate folding state after

de novosynthesis at the ribosome or proteins misfolded under stressful conditions are bound by Hsp70s in an Hdj-dependent manner and folded⁄ refolded to the native state Proteins that do not fold or that are diffi-cult to fold are released and rebound by Hsp70s sev-eral times (black symbols and arrows in Fig 8) In a stochastic process, CHIP associates with Hsp70–sub-strate complexes and recruits the E2 conjugating enzyme for ubiquitination of the substrate As Hsp70s are approximately 10–40 times more abundant than CHIP, only approximately 1–10% of the Hsp70– substrate complexes will be bound by CHIP with possible ubiquitination of the substrate Efficiently folding substrates (gray symbols and arrows in Fig 8) have only a small chance of being ubiquitinated Hsc70–CHIP complexes are less likely to bind

misfold-ed proteins The likelihood of at least one round of refolding is thereby increased This model of the triage decision allows sufficient time for refolding attempts

by the chaperones, keeping the amount of erroneously degraded chaperone substrates low Any increase in CHIP concentration due to physiological or patho-physiological processes will increase the clearance rate for damaged proteins, at an increased cost of degrad-ing proteins that are still useful

Experimental procedures Protein expression and purification

Human CHIP was produced in Escherichia coli and puri-fied by a combination of cation- and anion-exchange chromatography as described previously [2] Human Hdj1 and human Bag-1 were purified as described previously [37] Human Hop was purified from over-producing E coli strains as described previously [66,67] All proteins were quantified as described previously [68] using the Bio-Rad reagent (Bio-Rad Laboratories, Mu¨nchen, Germany) Human wild-type Hsp90b, Hsc70, Hsp70 and Aha1 were expressed with an Ulp1 cleavable N-terminal His6–Smt3 tag in E coli for 5 h at 30C (20 C for Aha1) and purified

as described previously [69] with some modifications The cells were lysed in a French press in 25 mm HEPES⁄ KOH

further purified on a Resource-Q column (GE Healthcare, Freiburg, Germany) Hsp90 and Aha1 were further purified

on a Superdex 200 Hiload 16⁄ 60 column (GE Healthcare)

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