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Despite this classical view, recent computational, structural and biochemical studies have highlighted the critical role of Keywords 5¢-deoxy-5¢-methylthioadenosine phosphorylase; CXC mo

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Archaea require a CXC motif for stability and folding

Giovanna Cacciapuoti, Iolanda Peluso, Francesca Fuccio and Marina Porcelli

Department of Biochemistry and Biophysics ‘F Cedrangolo’, Second University of Naples, Italy

Introduction

In the fascinating field of protein biochemistry,

ther-mostability and folding comprise important factors

that are currently gaining wide attention Disulfide

bonds represent an important structural feature of

many proteins, especially extracellular ones They not

only stabilize protein structures by lowering the

entropy of the unfolded polypeptide, but also are

required for the proper folding and biological activity

of several proteins Disulfide bond formation occurs in

the endoplasmic reticulum and mitochondrial

inter-membrane space of eukaryotes and in the periplasm of

prokaryotes [1] Disulfide bonds are a typical feature

of secretory proteins and are considered to contribute significantly to their overall stability [2] By contrast,

in intracellular proteins from well-known organisms, and as a result of the reductive chemical environment inside the cells [3], the presence of these covalent links

is limited to proteins involved in the mechanism of the response to redox stress [4] or to proteins catalyz-ing oxidation–reduction processes [1,5] Despite this classical view, recent computational, structural and biochemical studies have highlighted the critical role of

Keywords

5¢-deoxy-5¢-methylthioadenosine

phosphorylase; CXC motif and oxidative

protein folding; disulfide bonds;

hyperthermophilic proteins; purine

nucleoside phosphorylase

Correspondence

G Cacciapuoti, Dipartimento di Biochimica e

Biofisica ‘F Cedrangolo’, Seconda Universita`

di Napoli, Via Costantinopoli 16,

80138 Napoli, Italy

Fax: +39 081 5667519

Tel: +39 081 5667519

E-mail: giovanna.cacciapuoti@unina2.it

(Received 5 June 2009, revised 22 July

2009, accepted 29 July 2009)

doi:10.1111/j.1742-4658.2009.07247.x

5¢-Deoxy-5¢-methylthioadenosine phosphorylase II from Sulfolobus solfatari-cus (SsMTAPII) and purine nucleoside phosphorylase from Pyrococcus furiosus (PfPNP) are hyperthermophilic purine nucleoside phosphorylases stabilized by intrasubunit disulfide bonds In their C-terminus, both enzymes harbour a CXC motif analogous to the CXXC motif present at the active site of eukaryotic protein disulfide isomerase By monitoring the refolding of SsMTAPII, PfPNP and their mutants lacking the C-terminal cysteine pair after guanidine-induced unfolding, we demonstrated that the CXC motif is required for the folding of these enzymes Moreover, two synthesized CXC-containing peptides with the same amino acid sequences present in the C-terminal regions of SsMTAPII and PfPNP were found to act as in vitro catalysts of oxidative folding These small peptides are involved in the folding of partially refolded SsMTAPII, PfPNP and their CXC-lacking mutants, with a concomitant recovery of their catalytic activ-ity, thus indicating that the CXC motif is necessary to obtain complete reversibility from the unfolded state of the two proteins The two CXC-containing peptides are also able to reactivate scrambled RNaseA The data obtained in the present study represent the first example of how the CXC motif improves both stability and folding in hyperthermophilic proteins with disulfide bonds

Abbreviations

AdoMet, S-adenosylmethionine; GdnCl, guanidinium chloride; GSH, glutathione; GSSG, glutathione disulfide; MTA,

5¢-deoxy-5¢-methylthioadenosine; MTAP, 5¢-deoxy-5¢-methylthioadenosine phosphorylase; PDI, protein disulfide isomerase; PfCGC, NH 2 -RRCGCKD-COOH; PfPNP, purine nucleoside phosphorylase from Pyrococcus furiosus; PNP, purine nucleoside phosphorylase; sRNaseA, scrambled RNaseA; SsCSC, NH2-GSCSCCN-COOH; SsMTAPII, 5¢-deoxy-5¢-methylthioadenosine phosphorylase II from Sulfolobus solfataricus.

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these covalent links in the structural stabilization of

intracellular proteins in some hyperthermophilic

Archaeaand Bacteria [6–12]

The abundance of disulfides observed across

hyper-thermophilic organisms also has stimulated research

into the identification of the biochemical mechanisms

related to disulfide maintenance It was recently

dem-onstrated that specific protein disulfide

oxidoreducta-ses, which are structurally and functionally related to

protein disulfide isomerase (PDI) [13], play a key role

in intracellular disulfide shuffling in hyperthermophilic

proteins [14]

Two enzymes, 5¢-deoxy-5¢-methylthioadenosine

pho-sphorylase II from Sulfolobus solfataricus (SsMTAPII)

[10,11] and purine nucleoside phosphorylase from

Pyrococccus furiosus (PfPNP) [12], have been isolated

and characterized from hyperthermophilic Archaea

These enzymes are members of purine nucleoside

phos-phorylases (PNP), comprising ubiquitous enzymes of

purine metabolism that function in the salvage

path-way of the cells [15] SsMTAPII is a homohexamer

(subunit 30 kDa) characterized by extremely high

affinity towards 5¢-deoxy-5¢-methylthioadenosine

(MTA), a natural sulfur-containing nucleoside formed

by S-adenosylmethionine (AdoMet) mainly through

polyamine biosynthesis [16] SsMTAPII shares 51%

identity with human 5¢-deoxy-5¢-methylthioadenosine

phosphorylase (MTAP) and is able to recognize

adeno-sine [10], in contrast to human MTAP which is strictly

specific for MTA [17] PfPNP displays a much higher

similarity with MTAP than with PNP family members

[12] Similar to human PNP [18], PfPNP shows an

absolute specificity for inosine and guanosine [12]

SsMTAPII and PfPNP show features of exceptional

thermophilicity and thermostability and are

character-ized by stabilizing disulfide bonds Two pairs of

intra-subunit disulfide bridges have been revealed by the

crystal structure of SsMTAPII [11] and three pairs of

intrasubunit disulfide bridges have been assigned to

PfPNP by integrating biochemical methodologies with

MS [12]

It is interesting to note that SsMTAPII and PfPNP

contain, in their C-terminal region, an unusual CXC

motif (i.e cysteines separated by one neighbouring

amino acid X) as a typical feature, and that the

substi-tution of these two cysteines with serines significantly

affects both their thermodynamic and kinetic stability

[10,12], suggesting the involvement of the cysteine pair

in the thermal stabilization of both enzymes

In the present study, we demonstrate that the CXC

motif of SsMTAPII and PfPNP plays an important

functional role in the oxidative folding of these

enzymes and that two short CXC-containing peptides

with amino acid sequences corresponding to those present in the C-terminal region of SsMTAPII and PfPNP, respectively, act in vitro as functional mimics

of PDI The presence of a CXC motif in hyperthermo-philic proteins with multiple disulfide bonds, such as SsMTAPII and PfPNP, represents the first example of

a new molecular strategy adopted by these enzymes to improve their stability and to preserve their folded state under extreme conditions

Results and Discussion

SsMTAPII and PfPNP require a CXC motif to preserve their folded state

SsMTAPII and PfPNP are highly thermoactive, with

an optimum temperature of 120C, and extremely thermostable, with apparent Tm values of 112C and

110 C, respectively These enzymes are also character-ized by a remarkable kinetic stability and resistance to many chemicals, including SDS and guanidinium chlo-ride (GdnCl) [10,12] As demonstrated by structural and biochemical studies, SsMTAPII and PfPNP utilize multiple intrasubunit disulfide bridges as a principal mechanism to achieve superior levels of stability [11,12] A striking structural feature of these two enzymes is the presence in their C-terminal region of a cysteine pair, organized in an unusual CXC sequence motif This structural motif plays an important role in thermal stability because the substitution of cysteine with serine results in a significant lowering of the thermodynamic and kinetic stability parameters of SsMTAPII and PfPNP [10,12] The stabilizing role of the CXC disulfide appears almost intriguing Indeed, disulfide bonds are expected to make a higher contri-bution to thermostability when they join residues far apart in the primary structure Instead, in SsMTAPII and PfPNP, they are separated by a single residue of small size (i.e serine and glycine, respectively) Therefore, the stability imparted to the native protein structure could be almost marginal On the basis of these observations, it is possible to hypothesize that CXC plays an important functional role in stabilizing the protein through an oxidative folding mechanism involving the other structural disulfides of the enzyme Although rare in nature, few examples of oxidized CXC have been reported in the literature, including CSC in the Mengo virus coat protein [19], CSC in the L40C mutant of NADH peroxidase [20], CDC in AK1 protease from Bacillus sp [21], CTC in chaperone Hsp33 from Escherichia coli [22], CPC in redox-regu-lated import receptor Mia40 [23,24] and CGC in MTAP from P furiosus [9], which is highly

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homo-logous to SsMTAPII and PfPNP It is interesting to

note that a CGC motif at the C-terminus of the yeast

thiol oxidase Erv2p was found to be involved in the

exchange of the de novo synthesized disulfide bridge

with substrate protein [25] Moreover, it was

demon-strated that a synthesized CGC peptide and a CGC

motif in a mutant of E coli thioredoxin were

func-tional mimics of PDI [26] Taken together, these data

allowed us to speculate that the CXC motif in

SsM-TAPII and PfPNP could act as a redox reagent and

exert its stabilizing role by rescuing, in analogy with

PDI, any possible damage of the other disulfide bonds

of the protein To demonstrate the active role of CXC

motif in the oxidative folding process, we carried out

the unfolding of SsMTAPII, PfPNP and their

CXC-lacking mutants by incubation for 22 h at 25C with

6 m GdnCl in 20 mm Tris⁄ HCl (pH 7.4), containing

30 mm dithiothreitol The reversibility of the

GdnCl-induced unfolding was then examined by assaying

the catalytic activity after complete removal of the

denaturant

As shown in Fig 1, SsMTAPII and PfPNP are able

to refold with a recovery of catalytic activity of 59%

and 90%, respectively, compared to their control

enzymes By contrast, lower values of reactivation were

observed for SsMTAPIIC259S⁄ C261S and PfPNPC

254S⁄ C256S, the two CXC-lacking mutants (25% and 46% activity, respectively) These results demonstrate that the CXC motif is necessary to obtain almost com-plete reversibility from the unfolded state, and suggest that the cysteine pair is able to act as redox reagent in the rearrangement of scrambled disulfide bonds, thus contributing to the recovery of native and biologically active enzyme The observation that, in analogy with Erv2p [25], the CXC motif is part of a flexible C-termi-nal segment in SsMTAPII and in PfPNP [10,12], and that the CXC motif is very close to the two pairs of disulfide bridges in SsMTAPII [11], further strengthens this hypothesis

The data obtained in the present study represent the first example of how the CXC motif improves stability and folding in hyperthermophilic disulfide-containing proteins and could provide useful information with respect to engineering stable proteins and enzymes for therapeutic and industrial applications

From the sequence comparison of the C-terminal region of several PNPs present in databases, it appears that, despite their remarkable amino acid sequence identity, the CXC motif is conserved only in hyper-thermophilic enzymes, whereas it is absent in their mesophilic counterparts (Fig 2) This observation suggests a specific role of this structural motif in the stability of hyperthermophilic PNPs against extreme temperature and allows the hypothesis that the CXC disulfide could represent an additional aspect (i.e besides protein disulfide oxidoreductase protein) of the complex system involved in disulfide bond mainte-nance in hyperthermophilic organisms

NH2-GSCSCCN-COOH (SsCSC) and

NH2-RRCGCKD-COOH (PfCGC) act as in vitro catalysts of oxidative protein folding

PDI, the most efficient known catalyst of oxidative folding, is a multifunctional eukaryotic enzyme that utilizes the active site motif CGHC to catalyze the for-mation of native disulfides and the rearrangement of incorrect disulfide bonds, especially those within kineti-cally trapped, structured folding intermediates [13] In recent years, interest in protein folding in vitro has expanded rapidly, mainly focusing on the production,

in bacteria, of disulfide-containing proteins with poten-tial biotechnological applications Therefore, increased attention has been paid to the design and synthesis of novel, small-molecule reagents that could improve the efficiency of the oxidative folding process Recently, on the basis of the physical properties of PDI, a variety

of CXXC peptides have been synthesized and assayed [27] The active site of PDI has also been modeled as a

SsMTAPII

SsMTAPII C259S/C261S

PfPNP PfPNP C254S/C256S

100

60

80

SsMTAPII C259S/C261S PfPNP C254S/C256S

0

20

40

Fig 1 Refolding of SsMTAPII, PfPNP and their CXC-lacking

mutants after induced unfolding The reversibility of

Gdn-induced unfolding of MTAPII, PfPNP and their respective mutants

was started by a 20-fold dilution of the unfolding mixture and

extensive dialysis until complete removal of GdnCl Refolding was

analyzed by catalytic activity measurements performed under

stan-dard conditions U ⁄ R indicates SsMTAPII, PfPNP and their mutants

refolded after GdnCl-induced unfolding The activity of control

enzymes was expressed as 100% Each value is the average of

three separate experiments.

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CGC peptide, a molecule that, upon oxidation, forms

a strained 11-membered disulfide ring, representing a

good oxidizing agent [26] This peptide shows a

disul-fide reduction potential close to that of PDI and its

first thiol pKa is less than that of the natural redox

reagent glutathione Therefore, this CXC peptide is

able to function as an efficient catalyst of disulfide

isomerization [26] On the basis of these observations,

and in analogy with PDI, it is possible to hypothesize

a nucleophilic attack of the thiolate from CXC on an

incorrect protein disulfide followed by a thiol–disulfide

interchange within the substrate, leading in turn to a

native disulfide Two CXC-containing peptides, namely

SsCSC and PfCGC, whose amino acid sequences are

identical to those present in the C-terminal region of

SsMTAPII and PfPNP, respectively, have been

synthe-sized and their disulfide isomerase activity has been

assayed utilizing the partially refolded forms of

SsMTAPII, PfPNP and their CXC-lacking mutants as

substrates As shown in Fig 3, both peptides are

involved in the oxidative folding of these enzymes with

a concomitant recovery of their catalytic activity

Indeed, after 22 h of incubation in the presence of

SsCSC and PfCGC, the enzymatic activity of

SsMTAPII and its mutant, expressed as a percentage

of their control enzymes, reaches 86.8% and 51.8%, and 68% and 49.3%, respectively (Fig 3A) Similar results were obtained when the unfolded⁄ refolded forms of PfPNP and its mutant were assayed under the same experimental conditions (Fig 3B) It is inter-esting to note that, although PfPNP and its mutant show a higher reactivation values than SsMTAPII and its mutant (Fig 3), the ratio of these values with respect of their control enzymes is similar, thus indicat-ing that the efficiency of the process is comparable These data demonstrate that the CXC motif of SsM-TAPII and PfPNP is active, even when isolated from the proteins, and that it is able to induce the in vitro oxidative folding of these enzymes

To further confirm the ability of SsCSC and PfCGC

to function as efficient catalysts of oxidative protein folding, the disulfide isomerase activity of these CXC-containing peptides was assayed by monitoring the reactivation of scrambled RNaseA (sRNaseA) As shown in Fig 4, after 210 min of incubation in the presence of SsCSC and PfCGC, inactive sRNaseA shows a 5.5-fold and 3.6-fold activation, respectively, compared to the 8.9-fold activation observed in the presence of PDI These results demonstrate that the two CXC-containing peptides can act in the same way

Fig 2 Multiple sequence alignment of C-terminal regions of hyperthermophilic and mesophilic PNPs The CXC motif is shown in white lettering on a black background.

100

SsMTAPII C259S/C261S

PfPNP PfPNP C254S/C256S

20

40

60

80

0

U|R

U/R + PDI

U/R + SsCSC U/R + PfCGC

U|R U/R + PDI U/R + SsCS C

U/R + PfCGC

Fig 3 Effect of CXC-containing peptides on the reactivation of refolded SsMTAPII, PfPNP and their mutants SsMTAPII, PfPNP and their mutants, partially refolded after GdnCl-induced unfolding, (U ⁄ R), were incu-bated for 22 h at 30 C in the presence of various oxidative folding catalysts (reactiva-tion assay) The catalytic activity of (A) SsM-TAPII and SsMSsM-TAPIIC259S ⁄ C261S and (B) PfPNP and PfPNPC254S ⁄ C256S was then measured under standard assay conditions The activity of control enzymes was expressed as 100% Each value is the average of three separate experiments.

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as PDI, catalyzing the rearrangement of incorrect

disulfide bonds in a protein substrate It is interesting

to note that SsCSC displays a higher oxidative folding

activity than PfCGC (Figs 3 and 4), suggesting that

the presence in SsCSC of a third thiol in the sequence

CXCC could most likely enhance the reactivity toward

disulfide bonds

In conclusion, the results obtained in the present

study provide insight into the variety of molecular

mechanisms utilized for stabilizing folded proteins under

extreme thermal environments and allow us to speculate

that disulfide bonds and the CXC motif could combine

together to provide a novel stabilization strategy

Experimental procedures

Materials

Glutathione disulfide (GSSG), glutathione (GSH), bovine

liver PDI and bovine pancreatic sRNaseA were obtained

from Sigma GdnCl and dithiothreitol were obtained from

Applichem (Darmstadt, Germany) [methyl-14C]AdoMet

(50–60 mCiÆmmol)1 was supplied by the Radiochemical

Centre (Amersham Bioscience, Little Chalfont, UK) MTA

and 5¢-[methyl-14

C]MTA were prepared from unlabeled and

labeled AdoMet [10] Specifically synthesized

CXC-contain-ing peptides, PfCGC and SsCSC, were obtained from

PRIMM (Milan, Italy) All reagents were of the purest

commercial grade

Expression and purification of SsMTAPII, PfPNP

and their mutant forms

SsMTAPII, PfPNP and their CXC-lacking mutants (i.e

SsMTAPIIC259S⁄ C261S and PfPNPC254S ⁄ C256S) utilized

for these studies were expressed and purified as previously described [10,12] Manipulations of DNA and E coli were carried out using standard protocols [10,12,28] Protein concentration was determined by the Bradford assay [29]

Assays of enzyme activity

PNP activity was determined by monitoring the formation

of purine base from the corresponding nucleoside by HPLC using a Beckman System Gold apparatus (Beckman Coul-ter, Fullerton, CA, USA) The assay was carried out as described previously [12]

MTAP activity was determined by monitoring the forma-tion of [methyl-14C]5-methylthioribose-1-phosphate from 5¢-[methyl-14C]MTA [10] In all enzymatic assays, the amount of the protein was adjusted so that no more than 10% of the substrate was converted to product and the reaction rate was strictly linear as a function of time and protein concentration

GdnCl-induced unfolding and refolding

SsMTAPII, PfPNP and their respective CXC-lacking mutants (final concentration 0.4 mgÆmL)1) were incubated for 22 h at 25C in the presence of 6 m GdnCl in 20 mm Tris⁄ HCl (pH 7.4) containing 30 mm dithiothreitol Unfold-ing was probed by recordUnfold-ing the intrinsic fluorescence emis-sion To test the reversibility of the process, the refolding was started by a 20-fold dilution of the unfolding mixture

in Tris⁄ HCl 20 mm (pH 7.4) The refolded enzyme, after extensive dialysis against Tris⁄ HCl 20 mm (pH 7.4) until complete removal of GdnCl, was analyzed by catalytic activity measurements performed under standard condi-tions

Reactivation assay of SsMTAPII, PfPNP and their CXC-lacking mutants

The activity of SsCSC and PfCGC as catalysts of oxidative protein folding was tested by the ability to reactivate SsMTAPII, PfPNP, SsMTAPIIC259S⁄ C261S and PfPNPC254S⁄ C256S after GdnCl-induced unfolding and refolding

SsCSC and⁄ or PfCGC were first reduced with a five-fold excess of dithiothreitol in 50 mm Tris⁄ HCl (pH 7.4) for

10 min at 30C and then incubated at 30 C for 22 h in a reactivation mixture containing (in a final volume of

50 lL): 50 mm Tris⁄ HCL (pH 7.4), 2 mm EDTA, a gluta-thione redox buffer (1 mm GSH, 0.2 mm GSSG), 540 lm CXC peptide, and the protein to be reactivated (3 lg; final concentration 2 lm) After the incubation, SsMTAPII or PfPNP activity was measured under standard conditions The activity of control enzymes was expressed as 100% The positive control was represented by the reactivation of

0.8

0.4

0

Time (min)

Fig 4 Time course for the reactivation of sRNaseA by various

oxi-dative folding catalysts - - -, None; , PDI; d, SsCSC; , PfCGC.

Each value is the average of three separate experiments.

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the enzymes catalyzed by PDI (final concentration 0.11 lm)

under the same experimental conditions

Reactivation of sRNaseA

Disulfide isomerase activity was assayed as described

previ-ously [30] by monitoring the reactivation of sRNaseA, a

fully oxidized protein containing a random distribution of

its four disulfide bonds SsCSC and PfCGC were first

reduced with a five-fold excess of dithiothreitol in 50 mm

Tris⁄ HCl (pH 7.4) for 10 min at 30 C and then incubated

for 1 h at 30C in a reactivation mixture containing 0.1 m

Tris-acetate buffer (pH 8.0), 2 mm EDTA, a glutathione

redox buffer (1 mm GSH, 0.2 mm GSSG), 2 mm

CXC-pep-tide, and sRNaseA (0.5 mgÆmL)1in 10 mm acetic acid; final

concentration 19.6 lm) cCMP at a final concentration of

4 mm was then added and A296, as a result of

RNase-cata-lyzed cCMP hydrolysis, was monitored continuously for

210 min at 30C The positive control was the reactivation

of sRNaseA catalyzed by PDI (final concentration 4 lm)

The control for the non-enzymatic reactivation of sRNaseA

was represented by the same mixture without the addition

of any oxidative folding catalyst

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