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Báo cáo khoa học: Construction and biological activity of a full-length molecular clone of human Torque teno virus (TTV) genotype 6 pptx

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RNA transcription and DNA replication of this still poorly characterized ssDNA virus, we cloned the full-length genome of TTV genotype 6 and transfected it into cells of several types..

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molecular clone of human Torque teno virus (TTV)

genotype 6

Laura Kakkola1, Johanna Tommiska1, Linda C L Boele2, Simo Miettinen1, Tea Blom1,

Tuija Kekarainen2, Jianming Qiu3, David Pintel3, Rob C Hoeben2, Klaus Hedman1

and Maria So¨derlund-Venermo1

1 Department of Virology, Haartman Institute and Helsinki University Central Hospital, University of Helsinki, Finland

2 Department of Molecular Cell Biology, Leiden University Medical Center, the Netherlands

3 Department of Molecular Microbiology and Immunology, University of Missouri–Columbia, Life Sciences Center, Columbia, MO, USA

TT-virus (TTV) recently named Torque teno virus [1]

was found in 1997 in Japan from a patient with

post-transfusion hepatitis of unknown etiology [2] The

virus is non-enveloped and contains a single-stranded

circular DNA genome of approximately 3.8 kb [3,4]

To date, five major phylogenetic groups have been

defined [1] Due to its genome organization and

struc-ture, TTV resembles the chicken anemia virus (CAV)

of the Circoviridae family This family of veterinary

viruses comprises the genus Gyrovirus, including CAV,

and the genus Circovirus, including the porcine

circo-virus (PCV) and the beak and feather disease circo-virus of birds The human TT-virus is currently classified as a member of a new, floating genus, Anellovirus [1] The TTV genome consists of an approximately 2.6 kb coding and an approximately 1.2 kb noncoding region The latter contains a GC-rich area, a promoter and transcriptional enhancer elements [3,5–8] The transcriptional capacity of the minute viral genome is greatly expanded by splicing [9,10], resulting in six dis-tinct yet partially overlapping viral proteins [11] Little

is known of their functions However, the longest gene,

Keywords

Anellovirus; replication; Torque teno virus;

transcription

Correspondence

L Kakkola, Department of Virology,

Haartman Institute, Haartmaninkatu 3,

PO Box 21, University of Helsinki,

FIN-00014, Finland

Fax: +358 9 19126491

Tel: +358 9 19126676

E-mail: laura.kakkola@helsinki.fi

Website: http://www.hi.helsinki.fi/english

Note

Nucleotide sequence data are available in

the DDBJ ⁄ EMBL ⁄ GenBank databases under

the accession number AY666122

(Received 11 April 2007, revised 10 July

2007, accepted 11 July 2007)

doi:10.1111/j.1742-4658.2007.06020.x

Torque teno virus (TTV) is a non-enveloped human virus with a circular negative-sense (approximately 3800 nucleotides) ssDNA genome TTV resembles in genome organization the chicken anemia virus, the animal pathogen of the Circoviridae family, and is currently classified as a member

of a new, floating genus, Anellovirus Molecular and cell biological research

on TTV has been restricted by the lack of permissive cell lines and func-tional, replication-competent plasmid clones In order to examine the key biological activities (i.e RNA transcription and DNA replication) of this still poorly characterized ssDNA virus, we cloned the full-length genome of TTV genotype 6 and transfected it into cells of several types TTV mRNA transcription was detected by RT-PCR in all the cell types: KU812Ep6, Cos-1, 293, 293T, Chang liver, Huh7 and UT7⁄ Epo-S1 Replicating TTV DNA was detected in the latter five cell types by a DpnI-based restric-tion enzyme method coupled with Southern analysis, a novel approach to assess TTV DNA replication The replicating full-length clone, the cell lines found to support TTV replication, and the methods presented here will facilitate the elucidation of the molecular biology and the life cycle of this recently identified human virus

Abbreviations

CAV, chicken anemia virus; DIG, digoxigenin; PBMC, peripheral blood mononuclear cells; PCV, porcine circovirus; TTV, Torque teno virus.

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ORF1, is assumed to encode a capsid protein that may

also participate in DNA replication [3,12] as is the case

with CAV [13]

The infection mechanisms and pathogenicity of TTV

are unknown Putative replicative forms of TTV DNA

have been found in peripheral blood mononuclear cells

(PBMC), bone marrow and liver [14–16], suggesting

replication at these sites Low-level infectivity of TTV

has been shown in activated PBMC and in a few

human cell lines [17–19] The TTV promoter has been

shown to be active in both human [5,8,11] and

non-human cells [8,9] TTV mRNAs have been detected in

human PBMC [19], bone marrow [10] and several

other organs [20] However, the main host cells and

the target organs of this virus are still undefined

The study of the biological functions of TTV is

par-ticularly challenging Replication does not appear to

be very efficient in primary cells [17–19], nor in the few

cell lines supporting virus growth [17] On the other

hand, the veterinary circoviruses CAV and PCV have

been studied successfully with infectious plasmid clones

[21–24] With a full-length TTV plasmid clone of

geno-type 1, RNA transcription and splicing was studied in

Cos-1 cells However, neither DNA replication, nor

cell permissiveness was demonstrated [9]

The present study aimed to construct a full-length

TTV clone that can be used as a tool for exploring the

viral determinants important in virus–cell interactions,

and to find permissive cell lines for further molecular

and cell biological studies of this peculiar human virus

For this purpose, we have cloned and sequenced the

full-length genome of TTV genotype 6, and used it to

detect the key biological functions (i.e RNA

transcrip-tion and DNA replicatranscrip-tion) in a number of different

cell lines

Results

Genotype 6 cloning and sequence analysis

A full length molecular clone, pTTV, in plasmid

pST-Blue-1 was constructed of TTV genotype 6 (Fig 1A)

The cloned genome was sequenced (GenBank

acces-sion number AY666122; nucleotide numbering

accord-ingly), and found to be 3748 nucleotides in length A

TATA-box (TATAA) was located at nucleotides 83–87

and a poly(A) sequence ATTAAA at nucleotides

2978–2983 A GC-rich area was 107 nucleotides in

length In the present study, two forms of the

full-length clone were used for transfection experiments;

the excised linear genome (linTTV) and the intact

plas-mid pTTV (Fig 1B,C) The full-length plasplas-mid clone

contains at its left end the NG136 primer sequence [25]

in which two nucleotides differ from the in vivo geno-type 6 sequence However, the BspEI-excised linear construct excludes these primer-derived nucleotides

Production of TTV RNA Prior to the studies, all the cells were tested and found

to be TTV DNA negative by generic UTR-PCR and

by genotype 6 specific PCR

All seven cell lines were analyzed with RT-PCR and were found to produce identical TTV RNA upon transfection with either pTTV or linTTV In RT-PCR analysis of the TTV clone-transfected cells, two ampli-cons were observed (Fig 2): one from the spliced TTV mRNA (454 bp) and the other from TTV DNA (555 bp) RNase treatment prior to RT-PCR abolished the 454 bp amplicon; and DNase treatment abolished the 555 bp but not the 454 bp amplicon (Fig 2D) In addition, RT-PCR without the RT step, and RT-PCR

of the input DNA constructs, yielded only the 555 bp amplicon (Fig 2D) Furthermore, the sequence data of both amplicons showed that in the 454 bp amplicon the intron had, indeed, been spliced out These experi-ments substantiated that the 454 bp amplicon origi-nated from the transcribed viral RNA and not from DNA The TTV RNA was shown by RT-PCR to per-sist in subcultured cells for at least 11 days Nontrans-fected cells and cells transNontrans-fected with the backbone plasmid pSTBlue-1, remained negative for TTV RNA (Fig 2B,C) confirming the absence of endogenous, transcriptionally active TTV RT-PCR of retinoblas-toma mRNA yielded the expected amplicons (data not shown) demonstrating mRNA integrity The results were identical for all the cell lines

Replication of TTV DNA All seven cell lines were transfected with linTTV and with the intact pTTV For detection of TTV replication, total DNA from the transfected cells was treated with the restriction enzymes BamHI and DpnI, and subjected

to Southern analysis Two different probes were used (Fig 1): the one labelled with32P differentiates by size the replicating TTV DNA from the input; and the other labelled with digoxigenin (DIG) additionally documents the susceptibility of the input DNA to DpnI

In cells transfected with linTTV, the input DNA (after BamHI digestion) was seen with the DIG-probe as a 3004-bp fragment (Figs 1B and 3A,B, marked with filled circles), which was further digested by DpnI into a fragment of 2162 bp (Figs 1B and 3A,B, marked with filled squares) On day 3 post transfection, a full-length TTV DNA of 3748 bp (after BamHI digestion,

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detected with either probe) emerged in 293T, Huh7 and

UT7⁄ Epo-S1 cells, and less pronounced also in 293 and

Chang liver cells (Fig 3A,C, marked with an arrow)

However, no such bands appeared in KU812Ep6 and

Cos-1 cells That BamHI digestion yielded a full-length

fragment indicates that circularization of the input

lin-ear construct had occurred Furthermore, this 3748 bp

fragment was resistant to DpnI (Fig 3A,C, marked with

an arrow), indicating TTV DNA replication The

linear-ized backbone plasmid, not separated from the excised

linear TTV construct, did not replicate in these cells As

an additional specificity control for the DpnI assay,

three (HindIII, EcoNI or ScaI) restriction enzymes,

other than BamHI, were used in Southern analysis,

resulting in identical DpnI-resistant bands on day 3 The

DpnI-resistant DNA progressively accumulated in the

transfected cells from day 0 to day 3, as shown for 293T

cells in Fig 4 However, on days 3, 5 and 8–10 post

transfection upon cell passage, the amount of

DpnI-resistant (replicating) TTV DNA declined, and was

detectable in Southern analyses for up to day 5

Interest-ingly, in those cells that permitted replication of the

excised linear construct, high molecular weight double bands (sensitive to DpnI) were visible (Fig 3) Single-stranded DNA (ssDNA) could, however, not be visual-ized by Southern analysis, suggesting that its production remained below the detection limit Of note, when com-paring the 293 cells, with and without the SV40 large

T antigen, the amount of DpnI-resistant replicating TTV DNA detected on day 3 post transfection (relative

to a standard amount of total cellular DNA) was invari-ably much lower in 293 than in 293T cells (Fig 3A, and more pronounced in Fig 3C), suggesting a possible helper function for the SV40 large T antigen

In cells transfected with the intact pTTV, the input DNA (after BamHI digestion) was seen with the DIG-probe as a 3165 bp fragment (Figs 1C and 3A,B, marked with filled circles), which was further digested by DpnI into a fragment of 2162 bp (Figs 1C and 3A,B, marked with filled squares) On day 3 post transfection, the same 3165 bp fragment (Fig 3A, marked with an asterisk) was DpnI resis-tant, indicating replication of the complete pTTV The results with the 32P-probe verified this: the input

TTV genome

3748 bp

3004 bp

2162 bp

DIG

32 P

pSTBlue-1

3851 bp

32 P

2162 bp

3165 bp

DIG

32 P

32 P

pTTV total length

7775 bp

3748/1

TTV genotype6

NsiI

*

BspEI

pSTBlue-1

BspEI tscr tlat BspEI

TTVGCF NG136

A

Fig 1 The full-length clone and the

con-structs for transfection (A) The cloning

strategy of TTV into the pSTBlue-1 plasmid.

The GC-rich area is shown as a striped box

and the overlapping area in the clone as

spotted boxes The three products used in

the construction of the TTV clone are

indi-cated with black lines The key restriction

enzymes (see text for details), primers

(arrows: forward TTVGCF, reverse NG136),

the TATA-box (*), the poly(A) (d), the

tran-scription initiation (tscr) and the translation

initiation (tlat) sites are shown Schematic

representations of (B) linear BspEI-excised

construct (linTTV), and (C) pTTV construct

used in transfection experiments The viral

genome is represented by an empty bar and

the backbone plasmid by a thin black line.

The DIG- and the32P-labelled probes are

indicated BamHI (vertical bars) and DpnI

(r) restriction enzymes were used for the

analysis of DNA replication The predicted

TTV DNA products in replication analyses:

linTTV-derived 3004 bp fragment and

pTTV-derived 3165 bp fragment after BamHI

digestion are marked with d; linTTV and

pTTV derived 2162 bp fragments after

BamHI ⁄ DpnI-digestion are marked with j.

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pTTV was seen (after BamHI digestion) as two

DpnI-sensitive restriction fragments of 3165 bp and

4610 bp (Fig 3C) and, on day 3 post transfection,

these fragments had become DpnI resistant (Fig 3C)

In all pTTV-transfected cells, as detected with either

probe, a full-length DpnI-resistant 3748 bp fragment

(which would indicate rescue and replication of the

TTV genome from the backbone plasmid) remained absent

As opposed to the five other cell lines, in KU812Ep6 and Cos-1 cells, no replicating DNA (or, in some experiments with the latter cells, barely exceeding detection threshold) were detected upon transfection with either construct (Fig 3A) No apparent cyto-pathological changes were microscopically detected in the cells supporting TTV DNA replication

The results were the same regardless of the DNA iso-lation method (total cellular; Hirt extraction) and of the detection probes (DIG- and 32P-labelled probe) (data shown for 293 and 293T cells in Fig 3A,C) The non-transfected cells and the backbone plasmid-non-transfected cells were always negative for TTV DNA In Southern analysis, the input DNA served as an internal control to verify restriction enzyme activity: the input DNA was sensitive to DpnI (Fig 3A,B, marked with filled squares) whereas the newly synthesized DNA was resistant (Fig 3A, marked with an arrow) but remained digest-ible with other restriction enzymes (data not shown)

Circularization of the linear construct

In the linTTV-transfected cells on day 3, the emer-gence of the 3748 bp TTV DNA after BamHI diges-tion (Fig 3A) indicated that the input linear TTV DNA had circularized To confirm this, two other restriction enzymes (HindIII and ScaI, Fig 5A) that, like BamHI, cut the TTV genome only once, were used with identical results (i.e a single product of approximately 3.7 kb was detected; data not shown) The circularization was further confirmed by digestion

of the DNA samples with pairs of restriction enzymes that cut on both sides of the linearization breakpoint (Fig 5) BamHI⁄ SalI or XhoI ⁄ SalI double-digestions

of the input linear construct yielded three restriction fragments of approximately 2100, 740 and 890 bp with the first enzyme pair, and of 2200, 630 and 890 bp with the latter However, the same double-digestions

of replicating TTV DNA from 293T cells on day 3 post transfection yielded additional fragments of approximately 1630 bp and 1520 bp, respectively (Fig 5B), indicating fusion of the linearization break-point ends Taken together, these results show that, in the linTTV-transfected 293T cells, circular forms of the TTV genome had been formed

Effect of aphidicolin on TTV DNA replication

To reconfirm that TTV DNA replication had occurred and to investigate whether it utilizes the cellular replica-tion machinery, aphidicolin (an inhibitor of eukaryotic

C

B

D

DNA

nt 112 nt 182 nt 284

RT1F

nt 129-147

RT1R

nt 683-660

RNA

poly-A

D

non-transf.

cells Mw Mw

linTTV

500

400

300

bp

day3

Mw

pSTBlue-1 pTTV

Mw

500

400

600

day3

linTTV day3

400

500

600

400

500

600

input

linTTV

input pTTV +RT +RT

R

Fig 2 RT-PCR of TTV RNA (A) A schematic drawing of TTV

RT-PCR Transcription initiates at nucleotides 112, and splicing

removes nucleotides 182–284 [11] RT-PCR primers are shown

with arrows Amplicon sizes are 555 bp for DNA and 454 bp for

spliced mRNA RT-PCR results of representative 293T cells

trans-fected with (B) the linear excised TTV (linTTV) and (C) intact pTTV.

Nontransfected cells and cells transfected with the backbone

plas-mid (pSTBlue-1) were included as controls (D) RT-PCR controls

from 293T cells transfected with linTTV (day 3) and from the input

constructs +RT, normal RT-PCR; –RT, without the RT-step; R,

RNase-treated; D, DNase-treated.

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C

A

B

U bp 4899 3639 2799

Mw

input linTTV

U bp 3639 2799

Mw

input pTTV

4899

4899

3639

2799

293T

B B/D B B/D B B/D B B/D B B/D

bp

pSTBlue-1 Mw

293

B B/D B B/D B B/D B B/D B B/D

bp

pSTBlue-1

4899 3639 2799 Mw

4899 2799

Mw

Cos-1

KU812Ep6

B B/D B B/D B B/D B B/D B

cells

4899

3639

2799

Mw

3639

UT7/Epo-S1

cells

4899 2799 Mw

B B/D B B/D B B/D B B/D B B/D

bp

4899

3639

2799

Mw

Huh7

Chang liver

bp

4899

3639

2799

Mw

linTTV

input

bp 4625 3165 Fig 3 Southern analysis of TTV DNA replication (A) 293T, 293, KU812Ep6, UT ⁄ Epo-S1, Huh7, Cos-1 and Chang liver cells transfected with the excised linear (linTTV) or the intact pTTV construct The key products of the replication assay are marked in the 293T-cell figure: the input linTTV yielding a 3004 bp fragment and the input pTTV yielding a 3165 bp fragment after BamHI digestion are marked with d; the input linTTV and pTTV yielding 2162 bp fragments after BamHI⁄ DpnI digestion are marked with j; the DpnI-resistant circularized full-length TTV DNA is marked with an arrow; DpnI-resistant pTTV is marked with * (B) Southern analysis of the input constructs The products of the restriction enzyme digestions are marked as those in the 293T-cell figure (Note the absence of a 3748 bp product.) U, undigested (C) South-ern analysis of Hirt-extracted (BamHI ⁄ DpnI-digested) DNA from the 293T and 293 cells (with and without T antigen, respectively) transfected with pTTV or linTTV Input pTTV digested with BamHI as a control Arrows indicate DpnI-resistant full-length TTV DNA For detection of TTV DNA, either a DIG- (A,B) or a 32 P- (C) labelled probe was used.

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nuclear DNA replication) [26,27] was used The 293T

cells were transfected either with linTTV or with

pTTV, and were grown in the presence (versus

absence) of aphidicolin Upon aphidicolin treatment,

TTV DNA replication was blocked (Fig 6), indicating

nuclear DNA replication

Discussion Most of the data published on TTV are from PCR studies of clinical patient materials However, to apprehend the full impact of this human virus, more information on the molecular biology and host–cell interactions of this virus is needed To this end, we have cloned and sequenced the full-length genome of TTV genotype 6, and studied the key biological activi-ties of this virus The cloned viral genome was

3748 nucleotides in length; 105 nucleotides shorter than the TTV prototype TA278 (genotype 1, accession number AB017610) In genomic organization, geno-type 6 turned out to resemble the other known TTV types: a TATA-box and a poly(A) sequence flanking the coding region, and a GC-rich area of 107 bp located in the noncoding region

The biological activity of our full-length plasmid clone was analyzed in seven cell lines In a previous study, the TTV expression profile (i.e mRNAs tran-scribed and proteins translated) in 293 cells was deter-mined by using this full-length clone [11] In the present study, cells of all seven types, transfected with either pTTV or linTTV, produced TTV RNA The RNA transcripts were unequivocally documented with RT-PCR designed to flank a common splice site in the TTV genome In a previous study, TTV RNAs were

bp

4899

3639

2799

Mw

Fig 4 Accumulation of DpnI-resistant full-length TTV DNA (marked

with an arrow) in 293T cells from day 0 to day 3 post transfection.

293T cells were transfected with linTTV, and DIG-labelled probe

was used to detect the TTV DNA.

A

B

lin day3 Mw

BamHI/SalI

lin day3 Mw

XhoI/SalI

1953 1882 1515 1482

992

718

bp

2799 3639

H a B

I

la S

I

1631 bp

la S

I

1521 bp

744 bp

TTV

genome

3748 bp

SalI

H a B

I

o X

I

aI i d H

I

2227 bp

2117 bp

Fig 5 Circularization of the linear construct in 293T cells (A)

Sche-matic representation of the restriction enzyme cutting sites and the

corresponding restriction-fragment sizes (B) The input linear

con-struct (lin) and the total cellular DNA (from the linTTV-transfected

293T cells on day 3 post transfection) were digested with two

enzyme pairs (BamHI ⁄ SalI or XhoI ⁄ SalI), each pair cutting at both

sides of the linearization breakpoint Southern detection was

per-formed with a probe prepared by nick translation (the band

corre-sponding to the cloning vector pSTBlue-1 is indicated) The

fragments from the circularized TTV DNA are marked with arrows.

A

B

bp 4899 3639 2799

Mw

Fig 6 The effect of aphidicolin (APC) on TTV DNA replication 293T cells were transfected with pTTV or the linear (linTTV) con-struct in the absence (–APC) or presence (+APC) of aphidicolin (A) Southern analysis with the 32 P-probe of Hirt-extracted

(Bam-HI ⁄ DpnI-digested) DNA (B) Southern analysis with the DIG-labelled probe of BamHI- (B) and BamHI ⁄ DpnI-digested (B ⁄ D) DNA on day 3 post transfection Arrows are pointing to the sites of replicat-ing, DpnI-resistant TTV DNA.

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detected in Cos-1 cells transfected with a plasmid clone

of TTV genotype 1 [9] The cloning strategy used by

this group was similar to ours: the plasmid clone

con-tained overlapping genomic regions and the cloning

breakpoint was in the noncoding region Their

geno-type 1 clone and our genogeno-type 6 clone differed in the

lengths of the overlaps and in the locations of the

cloning breakpoints However, upon transfection, both

constructs produced RNA, indicating that cloning did

not impair the function of the promoter The promoter

area of TTV genotype 1 has been shown to be active

in many cell types, including Huh7 and cells of

ery-throid origin [5,8] Our results with genotype 6 were

similar, displaying TTV RNA expression (i.e promoter

activity) in diverse cell types

For the detection of replicating TTV DNA, we used

restriction enzyme analysis combined with Southern

hybridization This straightforward approach was

dem-onstrated to be useful for the study of TTV DNA

rep-lication TTV DNA replication was detected in the

majority of cell types When the excised linear

geno-type 6 DNA was transfected into 293T, 293, UT7⁄

Epo-S1, Chang liver and Huh7 cells, circularized and

replicating (DpnI resistant) forms of TTV DNA

accu-mulated up to days 3–4 It is possible that the DpnI

resistance might arise from replication-independent

demethylation of the input DNA, or from DNA

repli-cation not related to TTV However, both the input

linTTV and the cotransfected (together with linTTV)

backbone plasmid always remained sensitive to DpnI

This indicates that the DpnI-resistance of TTV DNA

was not due to replication-independent demethylation

of the input DNA, or due to general DNA replication,

but instead was a result of TTV DNA-specific

replica-tion That the production of the DpnI-resistant forms

could be abolished with a polymerase inhibitor further

verifies that replication had occurred within the cells

The high-molecular weight double bands that appeared

restrictively in the cells that supported TTV DNA

rep-lication (after transfection with linTTV), could

theoret-ically originate from concatamers formed from the

transfected DNA The bands were DpnI sensitive and

visible also in aphidicolin treated cells, suggesting that

they are formed without the cellular replication

machinery Whether these intracellular DNA forms are

required for initiation of replication remains to be

studied Of our seven cell lines, 293T showed the

high-est yield of replicating viral DNA, even exceeding that

of the Chang cells, which have been recently reported

to sustain TTV infection [17]

Upon transfection of five cell types with the intact

pTTV, DpnI-resistant (i.e replicating) DNA was

detected However, rescued forms of the TTV genome

were not seen, which could indicate inefficient excision from the backbone plasmid With other ssDNA-virus clones, the extent of rescue and replication seems to vary For example, a rodent parvovirus, HaPV, repli-cates and produces infectious virions when transfected

as an intact plasmid clone containing one copy of the viral genome [28] whereas, among the circoviruses, PCV gives a higher virus titer when cloned as tandem repeats [29] and CAV requires either tandem-repeat cloning or excision of the viral genome from the plas-mid before transfection [30] The rescue and replication

of the viral genomes could involve cellular and⁄ or viral proteins, with mechanisms possibly dependent on the particular virus (e.g nicking of DNA strands, recombi-nation, and rolling circle replication) [31,32] In com-parison with other ssDNA virus molecular clones, ours appear to resemble that of CAV; in order to produce genome-size replicating DNA, the viral genome needs

to be excised from the backbone plasmid before trans-fection

With PCV, it has been shown that the mere origin

of replication, cloned into the plasmid, can lead to replication of the entire plasmid, if transfected into PCV-infected cells [33] This raises the question whether replication of our pTTV (and maybe also linTTV) might have been assisted by coinfection with

a homologous virus However, all our cell lines were shown to be TTV DNA negative by generic PCR This suggests that the replicating TTV DNA in the present study was not produced with the help of endogenous TTV Interestingly, the amount of replicating TTV DNA was much lower in 293 than in 293T cells, suggesting that the SV40 large T antigen might provide some helper functions CAV and PCV have been shown to replicate efficiently in heterologously infected cells: CAV in MDCC-MSB-1 cells transformed with Marek’s virus [34] and PCV in pk-15 cells infected with swine papova virus [35–37] It is therefore possible that TTV indeed might benefit from some helper functions of other viruses for efficient produc-tive infection

By contrast to the other cell lines in the present study, the TTV clone showed little or no replication in KU812Ep6 and Cos-1 cells The former are of ery-throid origin, as are UT7⁄ Epo-S1, and could thus be thought to support TTV replication However, accord-ing to our unpublished data, the viability of KU812Ep6 cells declines during electroporation TTV,

a minute virus apparently lacking DNA polymerase, most likely depends on the cellular S-phase for replica-tion Indeed, our results with aphidicolin, which inhib-its eukaryotic nuclear DNA replication and blocks the cell cycle at early S-phase, strongly suggest that TTV

Trang 8

utilizes the cellular DNA polymerase and S-phase for

replication The requirement for rapid cell division,

together with the diminished viability of the

electropo-rated KU812Ep6 cells, could explain the lack of

repli-cation of our full-length clone in those cells The

deficiency of TTV replication in monkey

kidney-derived Cos-1 cells could represent species specificity

However, because mRNAs were produced, the

poten-tial species barrier does not appear to affect

transcrip-tion In previous studies, the sites of TTV replication

in humans have been suggested to be the

hematologi-cal compartment and the liver [15,16] Our results,

demonstrating replication of cloned TTV genotype 6

DNA in erythroid UT7⁄ Epo-S1, and hepatic Huh7

and Chang liver cells, further support this concept In

any case, the species and cell-type specificity of TTV

replication needs to be examined further The genetic

variation among TT viruses is extremely high To what

extent the various genotypes share biological functions

and possibly influence each others in coinfections

remains to be investigated At least the RNA

tran-scription of genotypes 1 and 6, which belong to the

same genogroup, appears to be similar [9,11]

The production of infectious virions from the

trans-fected cells could not be verified unequivocally with

the methods in use, and thus remains to be

investi-gated In our experiments, upon cell subculture, even

though the more sensitive RT-PCR continued to be

positive, the levels of replicating TTV DNA in

South-ern analysis declined below detection limit These

results suggest that, if infectious virions were

pro-duced, the infection did not spread efficiently to the

neighbouring cells, and⁄ or that the amounts of

prog-eny virions were relatively low It is possible that the

cells used in the present study did not express the

unknown TTV receptor, or that TTV simply does not

grow well in ordinary tissue culture, thereby

resem-bling many other viruses such as human

parvovi-rus B19, hepatitis C viparvovi-rus and human papillomaviparvovi-rus

A low infectivity of TTV is concordant with previous

studies showing that infection of Chang liver cells gave

rise to only low amounts of progeny virus [17] and

that, even in ex vivo PBMC cultures, the production of

TTV is scanty and requires cell activation [18,19]

Because TTV in vivo infects healthy individuals

chroni-cally, strict regulation of virus multiplicity and of cell

damage is mutually beneficial for the virus and its

host

The research on TTV-like animal circoviruses has

been greatly assisted by the availability of full-length

plasmid clones producing infectious virions in vitro

and clinical disease in vivo [21–24,29] Indeed, the

genetic map (defining viral mRNAs and proteins) of

TTV has been recently elucidated with this full-length TTV clone [11] In the present study, we demonstrated TTV-promoter activity in all cell types studied and, by a novel approach, identified five cell lines that supported TTV replication In the absence of knowledge on TTV proteins, replication and cell bio-logy, the full-length plasmid clone and replication assays presented here will be valuable tools for the examination of the mechanisms pertaining to the molecular biology, the cell tropism and the clinical sig-nificance of this recently discovered human virus

Experimental procedures

Cell lines and transfection methods

Seven cell lines, Cos-1, Huh7, Chang liver, KU812Ep6,

cells (African green monkey kidney; transformed with SV40) were maintained in DMEM containing 10% fetal

Invitro-gen, Carlsbad, CA, USA) and antibiotics The cells were transfected 1 day after subculture, at approximately 95% confluency, with 30 lL Lipofectamine2000 (Invitrogen Life Technologies, Carlsbad, CA, USA) and 5 lg DNA per

main-tained in MEM, with 10% fetal bovine serum and antibiot-ics, and were transfected as the Cos-1 cells Human liver cells, Chang liver (ECACC 88021102), were maintained as Huh7 cells, and transfected as Cos-1 cells (except that the amount of Lipofectamine2000 was 16 lL) Human kidney-derived 293T cells (expressing the SV40 T antigen,

were maintained in DMEM, 10% fetal bovine serum and antibiotics The 293T and 293 cells were transfected as the Cos-1 (except that the amount of Lipofectamine2000 was

25 lL); or by the calcium phosphate technique [40] The human erythroid leukemia cell line KU812Ep6 [41], kindly provided by Dr Miyagawa (Fujirebio Inc., Tokyo, Japan), was maintained in suspension in RPMI1640, 10% fetal

Janssen-Cilag, Berchem, Belgium) and antibiotics For transfection, the cells were harvested on days 3–4 For one reaction,

DNA was added The cells were electroporated immediately

at 300 or 350 V, and 960 lF (‘Gene Pulser’; Bio-Rad, Her-cules, CA, USA) A human erythroblastoid cell line

(To-hoku University School of Medicine, Japan), was main-tained in suspension in Iscove’s modified Dulbecco’s

erythropoietin and antibiotics For transfection, the cells

AMAXA Nucleofector system, using kit R and program

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T-24 (Amaxa Biosystems, Cologne, Germany) All the cells

PCR for the presence of endogenous TTV (generic

UTR-PCR, and genotype 6 specific PCR) [44]

Full-length cloning

The TTV genome was cloned in three overlapping fragments

(Fig 1A) The previously cloned 3.3 kb region of TTV

iso-late HEL32 [44] was completed to contain the full-length

gen-ome of the isolate The GC-rich part of the TTV gengen-ome was

amplified from the original serum [44] with seminested

prim-ers: forward TTVGCF (nucleotides 3206–3225) 5¢-CAGA

(nucleotides 216–199) 5¢-CGAATTGCCCCTTGACTG-3¢

and second reverse TTVGCR3 (nucleotides 157–140) 5¢-GG

GATCACCCTTCGAGGT-3¢ Both PCR reactions (volume

25 lL) contained 200 lm of each dNTP (Roche, Basel,

dimethylsulfoxide, 1 m betaine (Sigma, St Louis, MO, USA)

and 17.5 U of enzyme mix (‘Expand High Fidelity PCR

remain-ing 20 cycles The gel-purified PCR amplicon was cloned into

pSTBlue-1 AccepTor vector (Novagen, Madison, WI, USA)

in Escherichia coli DH5a cells

To facilitate the cloning of the entire TTV genome into a

single plasmid, a third overlapping piece of 396 bp was

amplified by PCR from the original serum with primers

GGGTTCCATT-3¢

Restriction enzyme digestions and ligations of the three

genomic parts resulted in a plasmid that contains the

entire TTV genome (pTTV), flanked by overlapping

175 bp areas, inserted between the EcoRI sites in the

pSTBlue-1 vector In addition, a BspEI restriction enzyme

can be used to excise from the TTV clone the complete,

single-unit (without overlaps) TTV genome in linear

form

Sequencing of the GC-rich region was carried out at the

DNA sequencing facility of the Institute of Biotechnology,

University of Helsinki, Finland All other sequencing

reac-tions in this study were done using the ABI Prism 3100

Genetic Analyzer (Applied Biosystems, Foster City, CA,

USA) in the sequencing core facility of the Haartman

Insti-tute, University of Helsinki, Finland

Plasmid constructs for transfection

For functional analysis of the TTV clone, the cells were

transfected with TTV plasmids of two different forms: an

uncut plasmid clone, pTTV, containing overlaps (Fig 1C),

and a linear construct, linTTV [full-length genome (without

overlaps) excised with BspEI, but not purified from the backbone plasmid; Fig 1B] The pSTBlue-1 backbone vector was used as a negative control The cells were collected for RNA and DNA analyses on days 1–11 post transfection

The transfection efficiencies were optimized with pEGFP-Luc vector (Clontech, Mountain View, CA, USA) encoding green fluorescent protein On day 1 post transfection, the percentage of green fluorescent protein-positive cells was estimated with a fluorescence microscope (‘Axioplan2’; Zeiss, Oberkochen, Germany)

RNA isolation and RT-PCR

Total RNA of the cells was extracted with TRIzol Reagent (Invitrogen Life Technologies) To remove residual DNA when necessary, the RNA samples were treated with DNase

RT-PCR was performed with a RobusT II RT-PCR Kit (Finnzymes, Espoo, Finland) in the presence of RNase inhibitor (‘RNaseOut’; Invitrogen Life Technologies, or

‘RNase Inhibitor’; Roche) For TTV-specific amplification, primers flanking the first common intron of 101 bp [11]

5¢-GCAGCGGCAGCACCTCGAA-3¢ and reverse RT1R (nucleotides 683–660) 5¢-GTCTAGCAGGTCCTCGTCTG CGAG-3¢ This separates the possible DNA-derived ampli-cons from the RNA-derived ones by size (555 bp and

454 bp, respectively; Fig 2A) The PCR program consisted

for 3 min RT-PCR for the cellular retinoblastoma mRNA was used as a control for RT-PCR and RNA isolation [45] RT-PCRs were carried out on DNase-treated and non-treated samples The RNA experiments were performed at least twice

DNA replication analyses

Total cellular DNA was isolated by cell lysis

Ontario, Canada) and by shearing through an 18G needle,

as previously described [28] The isolated DNA was extracted with phenol and chloroform, precipitated with ethanol and Na-acetate, and resuspended into water Total cellular DNA was also alternatively isolated with QIAamp DNA Blood Mini Kit (Qiagen, Hilden Germany) Low molecular weight DNA was alternatively extracted with the Hirt protocol [46]

The DNA samples (2.5–5 lg of Hirt-extracted or 30–

40 lg of total DNA) were digested with BamHI Subse-quently, half the digest was treated with DpnI (which cuts only prokaryotic DNA; New England BioLabs, Ipswich,

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MA, USA) The BamHI- and BamHI⁄ DpnI-digested DNA

samples were separated by gel electrophoresis The DNA

was transferred to nylon membranes (‘Hybond-N+’,

Amer-sham Biosciences, Piscataway, NJ, USA), and was

hybrid-ized overnight For hybridization, a DIG-labelled

TTV-specific probe, covering nucleotides 1803–2200 (Fig 1), was

prepared as described [44] using the outer primer pair of

genotype 6 PCR The DIG label was detected with

5-bromo-4-chloro-3-indolyl-phosphate (BCIP, Roche) and

4-nitro blue tetrazolium chloride (NBT, Roche) forming a

coloured precipitate For the circularization experiments,

another DIG-labelled probe was prepared to cover the

entire pTTV construct by DIG-Nick Translation Mix

consist-ing of a 560 bp fragment of the untranslated region

(NsiI-BspEI fragment from pTTV, Fig 1) was used The probe

the random primer method [47], and the signal was detected

by film exposure

(Sigma) was added to the medium, and 293T cells were

transfected by the calcium phosphate method The cells

were incubated in aphidicolin for 16 h, washed, and grown

further with aphidicolin On day 2 post transfection, the

DNA was isolated by the Hirt method, 10 lg of DNA was

were transfected with the Lipofectamine2000 method and

grown in the presence of aphidicolin On day 3 post

trans-fection, the total DNA was isolated, 30 lg of DNA was

hybridization with the DIG-probe

Acknowledgements

The authors thank Drs Ilkka Julkunen (National

Pub-lic Health Institute, Helsinki, Finland), Eiji Morita

and Kazuo Sugamura (Tohoku University School of

Medicine, Sendai, Japan), and Eiji Miyagawa

(Fujire-bio Inc., Tokyo, Japan) for cell lines, and Pa¨ivi Norja

and Heidi Bonde´n for excellent technical assistance

Dr Malcolm Richardson is acknowledged for revision

of the manuscript This study was supported by the

Finnish Technology Advancement Fund, the Finnish

Academy (project 76132), the Helsinki Biomedical

Graduate School, the Alfred Kordelin Foundation, the

Paulo Foundation, the Sigrid Juse´lius Foundation, the

Research and Science Foundation of Farmos, the Ella

and Georg Ehrnrooth Foundation, the Finnish

Kon-kordia Fund, the Helsinki University Central Hospital

Research and Education Fund, The Maud Kuistila

Memorial Foundation, The Finnish Foundation for

Research on Viral Diseases, and the Medical Society

of Finland (Finska La¨karesa¨llskapet)

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