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We subsequently investigated the biophysical features of HCV-1a and HCV-1b Core+1/S proteins using sequence analysis and complementary biophysical approaches [fluorescence, CD, dynamic li

Trang 1

ARFP/Core+1/S protein

Anissa Boumlic1,2,*, Yves Nomine´1,*, Sebastian Charbonnier1, Georgia Dalagiorgou2, Niki

Vassilaki2, Bruno Kieffer3, Gilles Trave´1, Penelope Mavromara2 and Georges Orfanoudakis1

1 Oncoproteins Group, Universite´ de Strasbourg, CNRS FRE 3211, Ecole Supe´rieure de Biotechnologie de Strasbourg, Illkirch, France

2 Molecular Virology Laboratory, Hellenic Pasteur Institute, Athens, Greece

3 Biomolecular NMR Group, UMR CNRS 7104, Institut de Ge´ne´tique et de Biologie Mole´culaire et Cellulaire, Illkirch, France

Introduction

Hepatitis C virus (HCV) is the major etiological agent

of chronic hepatitis, with more than 170 million people

being infected worldwide [1,2] Persistent HCV

infec-tion progresses, in 20% of cases, to liver cirrhosis

within 20 years of infection, with the possible

develop-ment of hepatocellular carcinoma (HCC) in 1–4% of

cases [3] No prophylactic vaccine against HCV exists,

and the efficiency of therapies is hindered by the

extreme heterogeneity of the HCV genome [4,5] HCV,

a Hepacivirus genus member of the Flaviviridae family,

is a small, enveloped RNA virus [6] Its genome is a positive, single-stranded 9.6 kb RNA containing 5¢-UTRs and 3¢-UTRs involved in viral protein trans-lation and viral replication [7–9] The genome encodes

a large precursor polyprotein that undergoes proteoly-sis, generating HCV structural proteins (Core, E1, and E2) and nonstructural proteins (p7, NS2, NS3, NS4A, NS4B, NS5A, and NS5B) An alternative reading frame (Core+1 ORF) overlapping the Core protein gene in the +1 frame was recently reported [10–13]

Keywords

ARFP/Core+1/S; hepatitis C virus (HCV);

intrinsic disorder; IUP/IDP; NMR

Correspondence

G Orfanoudakis, Oncoproteins Group,

Universite´ de Strasbourg, CNRS FRE 3211,

Ecole Supe´rieure de Biotechnologie de

Strasbourg, Illkirch, France

Fax: +33 3 68 85 47 70

Tel: +33 3 68 85 47 65

E-mail: georges.orfanoudakis@unistra.fr

*These authors contributed equally to this

work

(Received 25 October 2009, revised 30

November 2009, accepted 1 December

2009)

doi:10.1111/j.1742-4658.2009.07527.x

The hepatitis C virus (HCV) Core+1/S polypeptide, also known as alter-native reading frame protein (ARFP)/S, is an ARFP expressed from the Core coding region of the viral genome Core+1/S is expressed as a result

of internal initiation at AUG codons (85–87) located downstream of the polyprotein initiator codon, and corresponds to the C-terminal part of most ARFPs Core+1/S is a highly basic polypeptide, and its function still remains unclear In this work, untagged recombinant Core+1/S was expressed and purified from Escherichia coli in native conditions, and was shown to react with sera of HCV-positive patients We subsequently under-took the biochemical and biophysical characterization of Core+1/S The conformation and oligomeric state of Core+1/S were investigated using size exclusion chromatography, dynamic light scattering, fluorescence, CD, and NMR Consistent with sequence-based disorder predictions, Core+1/S lacks significant secondary structure in vitro, which might be relevant for the recognition of diverse molecular partners and/or for the assembly of Core+1/S This study is the first reported structural characterization of an HCV ARFP/Core+1 protein, and provides evidence that ARFP/Core+1/

S is highly disordered under native conditions, with a tendency for self-association

Abbreviations

ARFP, alternative reading frame protein; DLS, dynamic light scattering; HCC, hepatocellular carcinoma; HCV, hepatitis C virus; HSQC, heteronuclear single quantum coherence; IDP, intrinsically disordered protein; IMAC, immobilized metal ion affinity chromatography; MBP, maltose-binding protein; OG, n-octyl-b- D -glucoside; SSP, secondary structure propensity; TEV, tobacco etch virus.

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This ORF is responsible for the expression of various

alternative reading frame proteins (ARFPs), also

named Core+1 proteins, resulting from mechanisms

such as ribosomal frame shifting and internal initiation

at alternative AUG or non-AUG codons [10–12,14–

17] Core+1 proteins were recently shown not to be

required for HCV replication [18,19] However, the

presence of specific antibodies and T-cell-mediated

immune responses in serum from HCV-infected

patients suggests the expression of the Core+1 ORF

during HCV infection [10–12,20,21] Furthermore,

Core+1 proteins were found to interfere with

apopto-sis and cell cycle regulation [22,23], suggesting a

possi-ble role of these proteins in HCV pathogenesis

One remarkable ARFP is Core+1/S, a small

poly-peptide with a length varying from 38 to 76 residues

among HCV genotypes Core+1/S corresponds to the

C-terminal fragment of the Core+1 ORF, and to date

is the shortest ARFP form described Its translation

results from internal initiation at alternative AUG

codons (85–87) located downstream of the polyprotein

codon initiator Recently, two different groups

observed that Core+1/S is the predominant alternative

form when the Core+1 ORF is introduced into

mam-malian expression systems [16,24] In addition,

Core+1/S was found to be downregulated by the Core

protein and degraded in a proteasome-dependent

man-ner [25,26]

In order to further our understanding of these

pro-teins, we undertook biochemical and biophysical

stud-ies of the Core+1/S proteins derived from HCV-1a

and HCV-1b isolates The Core+1/S proteins were

produced in bacteria and purified in native conditions

ELISA experiments using the purified recombinant

Core+1/S of HCV-1b demonstrated the ability of the

protein to react with sera from HCV-infected patients

We subsequently investigated the biophysical features

of HCV-1a and HCV-1b Core+1/S proteins using

sequence analysis and complementary biophysical

approaches [fluorescence, CD, dynamic light scattering

(DLS), and NMR] We provide evidence that ARFP/

Core+1/S is highly disordered under native

condi-tions, with a tendency for self-association

Results

Sequence analysis of Core+1/S predicts the

largely disordered character of the protein

Sequence alignments were performed to analyze the

degree of Core+1/S amino acid conservation among

reference sequences of different HCV genotypes

(Fig 1A) [5] The N-terminal sequence is well conserved

and exhibits hydrophobic patches, encompassing resi-dues 1–6, 14–25, and 32–35 In contrast, considerable variability was observed in the location of the stop codon on the RNA sequence (data not shown), leading

to variation in the lengths of protein sequences Amino acid sequences were analyzed using the disorder predic-tion tools globplot and pondr globplot evaluates the sum of the disorder propensity for each amino acid among the sequence, and pondr analyzes the mean net charge and hydrophobicity of the polypeptide chain This combination of properties seems to be a prerequi-site for the absence of compact structure in native con-ditions [27] globplot predicted disordered regions encompassing amino acids 6–28 and 42–52 for HCV-1a Core+1/S, and amino acids 6–28 and 42–58 for HCV-1b Core+1/S, whereas pondr suggested that most of the Core+1/S sequence is disordered

In order to assess whether the disorder prediction is also confirmed by the absence of secondary structure, four algorithms (phd, gor4, simpa96, and sopma) were used to predict the secondary structure contents of both HCV-1a and HCV-1b Core+1/S proteins (Fig 1B)

A consensus is drawn for residues with at least three out of four identical secondary structure predictions Such a consensus suggested that the majority of resi-dues are not embedded in secondary structure elements, with the exception of short residue stretches mainly located in the second and third hydrophobic patches The combination of secondary structure and disordered region predictions strongly suggests that the N-terminal and C-terminal regions of HCV Core+1 proteins are largely unstructured and highly disordered (Fig 1A) These predictions are supported by the high degree of conservation of several disorder-promoting residues, such as alanines, arginines, glycines, and serines (Fig 1B) [28]

Expression and purification of Core+1/S proteins

in native conditions

We cloned and expressed the HCV-1a and HCV-1b Core+1/S proteins encompassing residues 85–160 and 85–142 of the full Core+1 ORF, respectively These constructs were fused to the C-terminus of either His6, His6–maltose-binding protein (MBP) (Fig S1), or His6–NusA (Fig 2A) Screenings of optimal yield and solubility conditions were first performed on the three constructs of HCV-1a Core+1/S by varying the induc-tion temperatures between 37 and 22C Analysis on Tris/Tricine SDS gels showed expression of proteins at the expected molecular mass (Fig 2B; Fig S1), with

an optimum induction temperature at 22C However, both His-tagged and MBP-tagged HCV-1a Core+1/S

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proteins were found largely in the insoluble fractions

after cell lysis (Fig S1), even after incubation at low

temperatures In contrast, NusA-tagged HCV-1a

Core+1/S was largely soluble even after tobacco etch

virus (TEV) protease cleavage (Fig 2C) The

solubiliz-ing properties of NusA have already been described in

the literature [29] However, it was surprising to

observe MBP fusion proteins in the insoluble fractions,

as the MBP carrier is also a well-known protein

solu-bilizer Despite its small size, Core+1/S seems,

there-fore, to promote aggregation of the fusion protein

when fused to the MBP carrier As NusA solubilized

HCV-1a Core+1/S, we fused the same carrier protein

to the HCV-1b Core+1/S After TEV protease-medi-ated proteolysis, both HCV Core+1/S proteins remained soluble (Fig 2C)

When Core+1/S production was scaled up, the use

of the optimal expression and purification conditions

as described above led to protein aggregation In order

to prevent this, we lowered the expression temperature

to 15C and systematically supplemented the purifica-tion buffer with l-arginine and l-glutamic acid at a final concentration of 50 mm each These additives are known to prevent protein aggregation [30] Finally,

A

B

Fig 1 Sequence analysis of Core+1/S proteins (A) Alignment of 17 Core+1/S amino acid reference sequences for different HCV genotypes Protein sequences were obtained after translation of the Core+1 ORF nucleotide sequences retrieved from the GenBank database (acces-sion numbers are given in parentheses) Core+1/S amino acid sequences were aligned using CLUSTALW Similarity percentages are indicated

on the right, according to CLUSTALW calculations Hydrophobic residues are boxed (B) Disorder and structure predictions Disorder predictions were made using GLOBPLOT and PONDR Disordered and ordered regions are indicated by ‘D’ and ‘.’, respectively Secondary structure predic-tions were performed with GOR 4, SOPMA , SIMPA 96 and PHD , using both HCV-1a and HCV-1b Core+1/S amino acid sequences as inputs (see Experimental procedures) Structure predictions for each residue position are indicated as a-helix (H), extended strand (E), b-turn (T), or ran-dom coil (C) Uppercase letters indicate a prediction rate higher than 80% A consensus was reported when three or more predictions over the four algorithms provide identical secondary structure prediction Residues are numbered from the start of Core+1/S and correspond to residues 85–161 and 85–144 of the Core+1 ORF, and nucleotides 599–827 and 599–776 of the Core/Core+1 RNA sequence, for HCV-1a and HCV-1b, respectively.

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buffers were routinely supplemented with dithiothreitol

and argon to reduce protein oxidation [31] Final

yields were approximately 1 mg of expressed protein

per liter of bacterial culture

Upon size exclusion chromatography, both HCV

Core+1/S proteins eluted as monomers, according to

column calibration (Fig 3A) MS analysis of the

puri-fied proteins gave experimental masses of 7630.7 ± 0.8

and 6076.0 ± 0.1 Da for HCV-1a and HCV-1b

Core+1/S, respectively The mass of HCV-1b Core+1/

S corresponds to the calculated value (6075.9 Da),

whereas that of HCV-1a Core+1/S showed loss of the

GA sequence that is usually left after TEV protease

pro-teolysis and the N-terminal methionine Purified

recom-binant Core+/1S proteins were also verified through

SDS/PAGE (Fig 3B), and were specifically recognized

by polyclonal antibodies against the Core+1 ORF in

western blot experiments (Fig 3C)

Sera from HCV-1-infected patients are reactive

against native HCV-1b Core+1/S

HCV-1b Core+1/S was used in ELISA to test the

reactivity of sera from patients positive for HCV

genotype 1 Figure 4 shows a high prevalence ( 60%)

of Core+1 antibodies in patient sera as compared with the cutoff value, defined as the average of the negative controls plus two standard deviations The presence of antibodies against Core+1/S indicates that the purified recombinant untagged protein remains immunoreac-tive, and suggests that the protein is present in patients infected with HCV of genotype 1

Intrinsic fluorescence of Core+1/S proteins HCV-1a and HCV-1b Core+1/S proteins contain tryptophans at positions 34, 49, 66, and 74, and posi-tions 6, 34, and 49, respectively Intrinsic fluorescence spectroscopy was therefore used to evaluate the solvent accessibility of these residues As all tryptophans are simultaneously excited, the emission spectrum results from the sum of the signals of individual emitters The maxima of fluorescence emission for HCV-1a and HCV-1b Core+1/S proteins were observed at wave-length of 354 and 353 nm, respectively (Fig 5A) These values are close to that of soluble tryptophan in aqueous solution (355 nm) [32], indicating that all try-ptophans of Core+1/S proteins are exposed to the

sol-A

B kDa

17 28

11

55 72

C

17 28

11

55 72

P S P S P S

TEV TEV

HCV-1a Core+1/S

Core+1/S NusA

T7

6xHis

TEV

17 28

11

55 72 NusA-HCV-1a Core+1/S

17 28

11

55 72

22 28

HCV-1a Core+1/S

NusA-HCV-1b Core+1/S

NusA-HCV-1b Core+1/S

HCV-1b Core+1/S

Fig 2 Expression and purification screenings of native NusA–HCV Core+1/S proteins (A) Cloning strategy for expression of Core+1/S The sequence His6–NusA is fused at the 5¢-terminus of the Core+1/S DNA sequence (B) Pellet/supernatant assays After transformation, expression of recombinant proteins was monitored for 2, 4 h or overnight at 37, 28 or 22 C, respectively Fifty microliters of bacterial cul-ture was sonicated and centrifuged for 15 min at 16 000 g Supernatants (S) and pellets (P) were analyzed by Tris/Tricine SDS/PAGE (C) IMAC purification of NusA–Core+1/S proteins followed by TEV protease digestion Labeled or unlabeled His6-NusA–Core+1/S proteins were expressed under optimized conditions, and purified on Ni2+–nitrilotriacetic acid resin in the presence of arginine and glutamic acid (50 m M each) After IMAC purification, fusion proteins were desalted and subjected to TEV protease cleavage to release Core+1/S proteins Lane 1: bacterial lysate Lane 2: IMAC elution at 250 m M imidazole Lane 3: desalted NusA–HCV-1a Core+1/S before TEV protease cleavage Lane 4: NusA–HCV-1a Core+1/S after TEV protease cleavage Lane 5: desalted NusA–HCV-1b Core+1/S before TEV protease cleavage Lane 6: NusA–HCV-1b Core+1/S after TEV protease cleavage Arrows on the right indicate the bands for soluble NusA-HCV-Core+1/S, NusA, TEV and Core+1/S proteins.

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vent In a second step, an HCV-1b Core+1/S sample

was subjected to a 20 min heat pulse 16 h prior to

flu-orescence analysis (Fig 5B) No change in either the

wavelength or the intensity of the maximum

fluores-cence emission was observed This observation

indi-cates an absence of precipitation, suggesting resistance

of the protein to heat treatment, a feature that is often

associated with disordered proteins [33]

Self-assembly of HCV Core+1/S proteins

DLS allows the oligomeric status of proteins in

solu-tion to be evaluated Hydrodynamic radius

distribu-tions were derived from DLS data recorded for each

protein sample under various conditions, assuming a

coil model as implemented in dynals (Fig 6) In the

absence of any treatment or additive (Fig 6, upper

panels), the average hydrodynamic radii (Rh) were

4.5 ± 2.4 and 2.5 ± 1.2 nm for purified HCV-1a and

HCV-1b Core+1/S proteins, respectively Assuming a

coil model, these radii are equivalent to particles of

nearly 15 and five monomers for 1a and

HCV-1b, respectively The radius distribution indicates the

polydisperse character of both isoforms As the

proteins eluted as monomers in a size exclusion chro-matography column, it appears that multimerization occurs during and/or after concentration

We previously showed that HCV-1a Core+1/S

is localized in the endoplasmic reticulum membranes [24] Under the hypothesis that HCV-1b Core+1/S contains membrane localization determinants, we added octyl glucoside [n-octyl-b-d-glucoside (OG)], a nonionic detergent that is frequently used to solubilize integral membrane proteins The presence of OG in Core+1/S proteins sharpened the size distributions as observed with DLS, and thus lowered the polydisper-sity in particle sizes, although the average hydrody-namic radii were not significantly altered (Fig 6, middle panels)

When the proteins were subjected to a heat pulse, the average hydrodynamic radii shifted from 4.5 ± 2.4

to 1.8 ± 0.6 nm for HCV-1a Core+1/S, and from 2.5 ± 1.2 to 1.6 ± 0.6 nm for HCV-1b Core+1/S (Fig 6, lower panels), suggesting a transition to lower-size oligomers In addition, the polydispersity significantly decreased Thus, high temperature is able

to disrupt Core+1/S multimers without leading to protein precipitation

12

8

4

20 40 60 80

0

Elution volume (mL)

6.5 13.7 29 43 67

100

28 55

11

c

kDa

kDa

A

C

(b)

(c)

HCV-1b Core+1/S

NusA

HCV-1a Core+1/S

28 55

11

28 55

11

11

kDa

17

28

Ponceau Red

Anti-Core+1 HCV-1a

Anti-Core+1 HCV-1b

Fig 3 Biochemical analysis of purified native HCV Core+1/S proteins (A) Size exclusion chromatography of HCV Core+1/S proteins After TEV protease proteolysis, proteins were injected onto a Hiload 16/60 Superdex 75 column in the presence of argi-nine and glutamic acid (50 m M each) The mass distribution in the eluant is indicated

at the top Both HCV-1a Core+1/S (dotted line) and HCV-1b Core+1/S (bold line) eluted

as monomers, according to the column cali-bration (B) Coomassie blue staining of puri-fied proteins by Tris/Tricine SDS/PAGE Molecular masses are given on the left, and arrows indicate the expected expression products (C) Western blot analysis of puri-fied Core+1/S proteins After purification and concentration, Core+1/S proteins were analysed by western blotting using anti-HCV-1a Core+1 or anti-HCV-1b serum Left panel: Ponceau staining of HCV Core+1/S proteins and HPV16 E6 Middle panel: HCV-1a Core+1/S revealed by anti-HCV-HCV-1a Core+1 serum Right panel: HCV-1b Core+1/S revealed by anti-HCV-1b Core+1 serum Molecular masses are indicated on the left.

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CD analysis of potential secondary structure of

Core+1/S proteins

CD spectra were recorded for both proteins in the

far-UV region Globally, CD spectra for HCV-1a

(Fig 7A) and HCV-1b (Fig 7B) Core+1/S proteins

did not show the characteristics of a full random coil

conformation (a strong negative minimum at 195–

198 nm, and a weak negative signal at 220 nm) [34]

Instead, we observed a maximum at 195 nm and a

minimum at 220 nm, suggesting the existence of

b-sheet secondary structure Deconvolution of the CD

data was performed using three sets of reference

pro-teins and the algorithms provided by the cdpro suite

[35] As selcon3 failed several times to fit the

CD data, this program was not used for data

analysis However, both cdsstr and contin/ll gave

consistent results, and allowed the contributions of

structural elements to be estimated The percentages

of a-helix (a), b-sheet (b) and unordered (U)

struc-tures were  5%,  30% and  65%, respectively,

with a typical range of variation of 10–20%

(Fig 7C) Although the high content of unordered

structure is consistent with disorder prediction, a sig-nificant amount of b-sheet content seems to be pres-ent The presence of such a signal might be due to the presence of intrinsic b-sheet structure in Core+1/

S protein Alternatively, it might also correspond to b-sheet structure formed at the interface of Core+1/

S monomers upon multimerization, as it has been shown that b-sheet structure is predominant in aggre-gates and is often associated with intrinsically disor-dered proteins [36]

Finally, the CD spectrum recorded for an HCV-1b Core+1/S sample subjected to a heat pulse was slightly different from that of an unheated sample (Fig 7D) In contrast, the addition of OG induced drastic changes in the CD spectrum as compared with the untreated sample spectrum for both Core+1/S proteins (Fig 7A,B), suggesting an effect of OG on the conformation of HCV-1b Core+1/S However, the addition of OG prevented the recording of data at wavelengths below 206 nm, hindering the deconvolu-tion of data

NMR analysis of HCV-1b Core+1/S

In order to further investigate the structural properties

of Core+1/S proteins, NMR 1H–15N heteronuclear single quantum coherence (HSQC) experiments were performed for both HCV-1b Core+1/S (Fig 8A) and HCV-1a Core+1/S (Fig S2) Both spectra exhibit a rather narrow amide proton chemical shift dispersion, limited to 0.7 p.p.m Such a range is characteristic of a lack of structural organization of the backbone [37] The spectrum recorded for HCV-1a Core+1/S showed

a high number of overlapping peaks, impeding the accurate counting of peaks In contrast, the HSQC spectrum of HCV-1b Core+1/S allows the counting of

a number of peaks consistent with that expected from the protein sequence

In order to assign backbone frequencies of the poly-peptide, three-dimensional NMR experiments were performed on a 15N,13C-labeled HCV-1b Core+1/S

0.4

0.0

0.3

0.2

0.1

HCV/HCC Controls

Fig 4 Reactivity of sera from genotype 1 HCV-infected patients

against HCV-1b Core+1/S The sera from HCV-infected patients

were tested by enzyme immunoassay, using the native HCV-1b

Core+1/S Controls correspond to HCV-negative patient sera.

was determined as the average of HCV-negative sera absorbance

plus two standard deviations.

305 325 345 365 385 400

Wavelength (nm)

Heated HCV-1a Core+1/S

0

6

HCV-1b Core+1/S

HCV-1b Core+1/S

305 325 345 365 385 400

Wavelength (nm)

0

6

3

Fig 5 Intrinsic fluorescence of Core+1/S

proteins UV fluorescence emission spectra

of Core+1/S proteins were recorded in

20 m M sodium phosphate buffer (pH 6.8,

2 m M ) (A) Fluorescence emission spectra of

HCV-1a and HCV-1b Core+1/S proteins in

buffer (B) Fluorescence emission spectra of

HCV-1b Core+1/S proteins were recorded

after boiling the protein for 20 min and

cool-ing to room temperature.

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sample Near-complete1HN,15N-backbone and13

C-res-onance assignment could be achieved for HCV-1b

Core+1/S (Fig 8A), with the exception of His14 and

Ser38, as well as the first two residues (Gly-Ala)

remaining from the TEV protease site The lack of

His14 resonances might be due to

protonation–depro-tonation equilibrium of the imidazole ring [38] The

absence of Ser38 resonances needs to be further

inves-tigated We used experimental carbon chemical shifts

to probe the presence of helical or b-sheet secondary

structures For all residues of HCV-1b Core+1/S, Ca

secondary chemical shifts were below 1.0 p.p.m

(posi-tive or nega(posi-tive) (Fig 8B), confirming the absence of

stable secondary structure elements in HCV-1b

Core+1/S However, a consensus was observed for

residues encompassing the region between residues 32

and 35, suggesting that this region might have a

ten-dency to b-sheet character Interestingly, the same

region was predicted to contain b-sheet elements by

the majority of the secondary structure prediction

methods, and also corresponds to a nondisordered

region according to globplot analysis (Fig 1B)

Methods based on chemical shifts are often used to

depict secondary structure elements, but quantitative

interpretation of secondary chemical shifts alone

remains difficult, because the expected values for fully

formed secondary structures vary for different amino

acids [39] In order to quickly visualize the fractional

deviation of the experimental chemical shifts from pure

a-helix or b-sheet secondary shifts, residue-specific

sec-ondary structure propensity (SSP) scores of HCV-1b

Core+1/S were calculated on the basis of ssp software

recommendations [40] ssp combines chemical shifts

from different nuclei weighted by their sensitivity to a-helix or b-sheet structures into a single SSP score varying between 0 and 1, or 0 and )1, for a-helix and b-sheet structures, respectively These scores represent the expected fraction of a-helix or b-sheet secondary structure for each residue Calculated scores of HCV-1b Core+1/S are very close to zero values, indicating

an overall low SSP In particular, the SSP profile shows almost no propensity to adopt a helical confor-mation along the protein sequence Although a mild propensity to adopt a b-sheet conformation is visible for residues encompassing the regions between 3 and

8, 32 and 35, and 41 and 44, it is very limited as compared to the maximal amplitude expected for a full b-sheet conformation

Finally, the1H–15N-HSQC NMR spectrum recorded for HCV-1b Core+1/S in the presence of 6% OG (Fig 8D) showed a few notable changes for Val21, Ile33, Trp34, Val35, Thr47, and five glycines distrib-uted all over the sequence (Gly7, Gly8, Gly22, Gly30, and Gly50) These results suggest a possible weak interaction of HCV-1b Core+1/S with OG

Discussion

HCV Core+1/S proteins are intrinsically disordered Core+1/S proteins correspond to the C-terminal parts

of most of the described HCV ARFPS To date, nei-ther biochemical nor biophysical data have been described for ARFPs Here, we succeeded in producing the Core+1/S proteins from HCV-1a and HCV-1b genotypes, using the standard Escherichia coli BL21

Hydrodynamic radius (nm) Hydrodynamic radius (nm)

0.0

0.8

0.4 0.0

0.4

0.2

0.8

0.4

R ave : 2.5 nm s: 1.2 nm

R ave : 2.6 nm s: n/a

R ave : 1.6 nm s: 0.6 nm

R ave : 4.5 nm s: 2.4 nm

R ave : 4.0 nm s: n/a

R ave : 1.8 nm s: 0.6 nm

0.0

10.0 8.0 6.0 4.0 2.0 0.0 0.0

0.4

0.2

10.0 8.0 6.0 4.0

2.0

0.8

0.4

0.0

0.8

0.4

HCV-1a Core+1/S HCV-1b Core+1/S

control

OG

Heat pulse

Fig 6 Size distribution histograms of HCV-1a and HCV-1b Core+1/S proteins deter-mined by DLS Twenty microliters of 80–100 l M protein samples in 20 m M sodium phosphate buffer (pH 6.8, 400 m M NaCl) were directly analyzed, incubated with

OG, or subjected to a heat pulse prior to analysis Samples were analyzed by DLS, and the hydrodynamic radius distributions of Core+1 proteins were determined using DYNALS , assuming a coil model Solid lines are the three-parameter nonlinear least squares fits of the size distribution profiles using a Gaussian model, yielding average radii (R ave ) and widths at the half-height (s) When the profile exhibits only two values,

an average radius was determined by weight averaging of the intensities.

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bacterial system We optimized the expression and

purification processes under native conditions, and

obtained substantial amount of native, highly pure,

untagged proteins We detected antibodies against recombinant 1b Core+1/S in the sera of HCV-infected patients, suggesting that the protein might be expressed during HCV infection, either alone or as a part of a larger ARFP

Combining the results of complementary biophysical techniques, our study showed that Core+1/S proteins lack secondary and tertiary structure 1H–15N-HSQC NMR experiments performed on both HCV-1a and HCV-1b Core+1/S constructs showed a limited chemi-cal shift dispersion of amide proton resonances into a narrow range (0.7 p.p.m) This is indicative of a disor-dered state, as inherent flexibility and rapid intercon-version between multiple conformations generally lead

to a poor chemical shift dispersion Exceptions are the

15N-backbone resonances in 1H–15N-HSQC spectra of Core+1/S proteins These resonances are influenced both by residue type and by the local amino acid sequence, and therefore remain well dispersed, even in fully unfolded states [41] In addition, the distribution

of correlation peaks around 10 p.p.m in the HSQC spectrum, which are assigned to tryptophan side chains, indicates that these residues lie in a very similar environment, in agreement with fluorescence data indicative of solvent-exposed tryptophans Together with the absence of consensus in the backbone carbon chemical shift differences, these observations suggest a lack of secondary structure for HCV-1b Core+1/S This conclusion is further reinforced by the high con-tent of unordered conformation ( 65%) determined

by CD spectroscopy Finally, the HSQC spectrum recorded for HCV-1a Core+1/S also displays a poor proton chemical shift distribution, suggesting that this protein is also disordered

When subjected to a heat pulse, folded proteins commonly unfold and precipitate, owing to solvent exposure of hydrophobic residues, whereas nonfolded peptides may remain in solution [33] We demonstrated that HCV-1b Core+1/S remains soluble after heat pulse treatment, as observed on fluorescence spectra Moreover, DLS shows that the mass distribution shifts

to lower molecular masses This is confirmed by the observation in NMR spectra of more intense peaks following a heat pulse (data not shown) No significant change was observed in CD spectra after such treat-ment, indicating that this treatment does not influence the global conformation of the polypeptide

Intrinsically disordered proteins (IDPs) are defined

as proteins containing at least one disordered region, and were recently recognized as a new protein class [42] Disordered proteins are gaining considerable attention, owing to their capacity to perform numer-ous biological functions despite their lack of defined

Wavelength (nm)

Wavelength (nm)

buffer OG

10

15

buffer OG

HCV-1a Core+1/S

HCV-1b Core+1/S

0 50 100

C

U

0

–5

–10

–15

–25

5

–20

A

B

10

15

0

–5

–10

–15

–25

5

–20

α β

θ[MR

2·dmol

θ[MR

2·dmol

Fig 7 Far-UV CD analysis of HCV Core+1/S proteins Data are

rep-resented as molar ellipticity per residue Core+1/S proteins (4 l M )

in 20 m M sodium phosphate buffer, 50 m M NaCl, and 0.15 m M

dith-iothreitol (A, B) CD spectra of HCV-1a and HCV-1b Core+1/S

pro-teins in buffer (solid line), after incubation with 6% OG (C) Far-UV

data were analyzed with the CDP ro package, using two algorithms

( CONTINLL , and CDSSR ) and three protein databases (SP43, SMP56,

and SDP48) a, a-helix; b, b-sheet; U, turns and unordered

second-ary structure.

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structure [42–47] Under native conditions, Core+1/S

proteins remain unstructured, and should therefore be

classed as IDPs This character is also confirmed by

disorder and structure predictions based on protein

sequences This is not the first time that an HCV

protein has been reported to be at least partially

disordered Indeed, the first 82 amino acids of the

N-terminal part of Core protein and domain 2

of NS5A protein have already been classed as IDPs

[48–50] Domain 3 of NS5A is also natively unfolded

[51] More generally, intrinsic disorder is commonly

found in viruses For instance, among Flaviviridae,

Dengue virus, West Nile virus and bovine viral

diar-rhea virus capsid proteins contain flexible, basic

regions [52–54] Proteins from other virus families

were also identified as being partially or completely

disordered, such as the Nef protein of simian immu-nodeficiency virus [55], HIV tat protein [56], and the nucleoprotein and phosphoprotein of the measles virus [57,58] As virus genomes are restricted in molecular size, the flexible nature of disordered regions of pro-teins may allow efficient interaction with several tar-gets [59]

HCV Core+1/S proteins tend to self-associate The deconvolution of Core+1/S CD spectra suggested the presence of a significant proportion of b-sheet sec-ondary structures (30%), in disagreement with the NMR-derived SSP A first hypothesis to explain this

is the difference in concentration range used to obtain CD and NMR data However, the position and

Cα

Cβ

C0

Amino acid sequence

B

–1

0

1

2

–2

–1

0

1

2

–2

–1

0

1

2

–2

Without OG

With OG 6%

V21

I33 V35

W34 A17

G30

G22

G7 T47 G50

G8

8.0 8.2

114

118

122

126 110

130

A17

A56

W34

I33 A2

A5

R36

A43

R42R4

A44

R53

I39

V32

F52

W49 V35

A23 C13 S3 L20 R31 W6 V21

V29 V16

M3

Q9 T26

S55

D10

M25

M1

S48 S54 S45

S12

S41

G22

T51 T47 G50

G28

G7

G30

G8

1 H (p.p.m.)

8.0 8.2

10.0 130

Amino acid sequence

–0.5

0.5 1.0

–1.0 0.0

C

Fig 8 NMR results for HCV-1b Core+1/S (A) Standard 2D 1 H– 15 N-HSQC spectrum recorded at 600 MHz and 22 C on a

100 l M sample of HCV-1b Core+1/S Each cross-peak corresponds to a correlation between an amide hydrogen atom and a nitrogen atom Assignments have been deposited in the BMRB (Ref 16487) (B) Differences between experimental carbon chemical shifts and random coil values as a function of sequence number (C) SSP of HCV-1b Core+1/S Carbon chemical shifts were used to calculate the residue-specific SSP scores of HCV-1b Core+1/S by follow-ing the SSP software recommendations Positive values ranging from 0 to 1 and neg-ative values ranging from 0 to )1 represent the propensities to form pure a-helix and b-sheet structures, respectively (D) Effects

of the nonionic detergent OG on HCV-1b Core+1/S The superimposition of 2D

1 H– 15 N-HSQC spectra of HCV-1b Core+1/S

in the absence (blue) or presence (green) of 6% OG is shown.

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bandwidth of peaks from HSQC spectra recorded with

30 or 400 lm HCV-1b Core+1/S samples are strictly

identical (data not shown), suggesting the absence of a

concentration effect, at least in this concentration

range On the other hand, the fact that the NMR

tech-nique is a very powerful method, allowing recording of

data at an atomic level, raises the question of potential

problems with experimental CD data collection and/or

inappropriate reference databases used to fit the CD

data First, CD data were collected and analyzed

fol-lowing the key considerations well described by

Green-field [60], allowing us to reasonably rule out data

collection issues, although they are not fully excluded

Second, the reference databases are derived from

glob-ular soluble proteins, and include only a few

disor-dered proteins For instance, the SDP48 reference

database employed in the present study contains only

five denaturated proteins in a total of 48 proteins

Therefore, the use of these databases for nonglobular

proteins is not really appropriate, as peptides or

disor-dered proteins tend to adopt multiple conformations in

equilibrium rather than a single structure

Although the CD results might overestimate the

b-sheet content, both CD and NMR data qualitatively

indicate a b-sheet secondary structure propensity This

observation suggests that the detected b-sheet signal

could be due to partial oligomerization of the natively

disordered HCV Core+1/S proteins This hypothesis

is also supported by the DLS results, which reveal the

existence of relatively high molecular mass particles in

protein samples, although previously purified in a

monomeric form by size exclusion chormatography

The residues involved in such oligomerization might be

located in the core of the protein between Ile33 and

Val35, as suggested by the chemical shift deviations

from random coil values Despite their lack of

folded and globular structure, intrinsically disordered

states of proteins often possess significant amounts of

transient structure [47]

Biological roles of Core+1/S proteins

Most HCV proteins contain membrane anchor

domains [61] The presence of hydrophobic patches on

Core+1/S protein sequences supports the hypothesis

that the proteins might contain membrane association

determinants, which may partially explain the

polydis-perse behavior of the protein in aqueous solution

Interestingly, confocal microscopy and Triton X-100

cell fractionation have previously demonstrated that

HCV-1a Core+1/S localizes in internal membranes

and the endoplasmic reticulum of transiently

transfect-ed Huh7 cells [25] Furthermore, the Core protein itself

has been found to be associated with membranes [48] The influence on Core+1/S behavior of OG, a non-ionic detergent known to solubilize integral membrane proteins, was therefore investigated further DLS showed that OG micelles reduce Core+1/S dispersity Moreover, the CD spectrum showed a change on the addition of the detergent Finally, HSQC experiments showed that only a few residues are affected by the presence of OG Taken together, these results are indicative of a possible weak interaction with the detergent, as is often observed for IDPs However, further experimental data on the structural character-ization of a putative interaction between Core+1/S proteins and membranes and comparison with the membrane association properties of the Core protein would be required

The presence of circulating antibodies against the HCV Core+1/S proteins suggests that their expression might occur at a certain stage of HCV infection Furthermore, the facts that Core+1/S proteins are disordered under native conditions, and that their ORFs are well conserved among HCV genotypes, support the hypothesis that the disordered nature of Core+1/S proteins might have some roles during HCV infection The disordered nature of the Core+1/

S proteins, which confers conformational and recogni-tion plasticity to the proteins, may be required for the binding of different partners through the same region,

as is typical for natively disordered proteins [59] This feature is often found for proteins involved in cell signaling and regulation [44] Our study contributes to the characterization of the Core+1/S proteins, provid-ing new insights into their biophysical properties Further studies will be required to identify the cellular targets of Core+1/S proteins, enabling the characteri-zation of the role of Core+1/S proteins in HCV pathogenicity

Experimental procedures

Protein sequence analysis

To analyze the degree of conservation of the Core+1/S amino acid sequence among HCV genotypes, Core+1/S amino acid sequences were deduced from the Core+1 ORFs of different HCV genotypes retrieved from the NCBI website (http://www.ncbi.nlm.nih.gov) [5] and aligned using

was performed using globplot [63] and pondr [64] Sec-ondary structure predictions were performed on HCV-1a and HCV-1b Core+1/S, using four algorithms (sopma,

website (http://pbil.ibcp.fr/)

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