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Enhancement of intracellular concentration and biological activity of PNA after conjugation with a cell-penetrating synthetic model peptide Johannes Oehlke1, Gerd Wallukat2, Yvonne Wolf1

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Enhancement of intracellular concentration and biological activity of PNA after conjugation with a cell-penetrating synthetic model peptide

Johannes Oehlke1, Gerd Wallukat2, Yvonne Wolf1, Angelika Ehrlich1, Burkhard Wiesner1, Hartmut Berger1 and Michael Bienert1

1

Institute of Molecular Pharmacology, Berlin, Germany;2Max Delbru¨ck Center for Molecular Medicine, Berlin, Germany

In order to evaluate the ability of the cell-penetrating

a-helical amphipathic model peptide KLALKLALKALK

AALKLA-NH2 (MAP) to deliver peptide nucleic acids

(PNAs) into mammalian cells, MAP was covalently linked

to the 12-mer PNA 5¢-GGAGCAGGAAAG-3¢ directed

against the mRNA of the nociceptin/orphanin FQ receptor

The cellular uptake of both the naked PNA and its

MAP-conjugate was studied by means of capillary electrophoresis

combined with laser-induced fluorescence detection,

confo-cal laser scanning microscopy and fluorescence-activated cell

sorting Incubation with the fluorescein-labelled

PNA–pep-tide conjugate led to three- and eightfold higher intracellular

concentrations in neonatal rat cardiomyocytes and CHO

cells, respectively, than found after exposure of the cells to the naked PNA Correspondingly, pretreatment of sponta-neously-beating neonatal rat cardiomyocytes with the PNA–peptide conjugate and the naked PNA slowed down the positive chronotropic effect elicited by the neuropeptide nociceptin by 10- and twofold, respectively The main rea-sons for the higher bioavailability of the PNA–peptide conjugate were found to be a more rapid cellular uptake in combination with a lowered re-export and resistance against influences of serum

Keywords: cell-penetrating peptides; cellular uptake; PNA– peptide conjugates

The wider application of peptide nucleic acids (PNAs) [1] as

antisense agents appears to be limited mainly by poor

cellular uptake [2,3] Improved delivery into mammalian

cells and enhanced antisense activity have been achieved

after covalent coupling of PNAs to cell-penetrating peptides

(CPPs), which are able to enter cells in a nonendocytic but

as yet unknown mode [3–8] The structural requirements for

the delivery activity of peptides have been unclear until now

In order to contribute to an elucidation of structure–

delivery activity relationships we have previously

investi-gated the cellular uptake and biological activity of

CPP–phosphorothioate oligonucleotide conjugates using

the cell-penetrating amphipathic model peptide MAP

(KLALKLALKALKAALKLA-NH2) [9, 10] as the lead

compound [11] The value of the results of this study was

limited, however, by a high cell toxicity of the

phosphoro-thioate oligonucleotide–peptide conjugates Therefore, in

the present study we evaluated the suitability of PNA to

serve as the cargo molecule in MAP-based structure– delivery activity investigations To this end we investigated cellular uptake and biological activity of a 12-mer peptide nucleic acid (5¢-GGAGCAGGAAAG-Lys-3¢; compound I; Table 1) complementary to bases 12–23 of the translated region of the nociceptin/orphanin FQ receptor, proven previously to be sensitive to antisense attacks [12,13], and of its conjugate with MAP (compound II; Table 1) For assessing the cellular uptake, we developed a protocol based

on capillary electrophoresis with laser-induced fluorescence detection (CE-LIF) providing absolute quantities of inter-nalized PNA which was used supplementally with confocal laser scanning microscopy (CLSM) and fluorescence-acti-vated cell sorting (FACS)

Material and methods General

Chemicals and reagents were purchased from Sigma (Deisenhofen, Germany), Bachem (Heidelberg, Germany)

or PE Biosystems unless specified otherwise Release of lactate dehydrogenase was assessed by means of LDH-L reagent from Sigma

Synthesis of PNA and PNA–MAP conjugates PNA oligomers were synthesized manually using the t-Boc strategy [14] The peptide segments of the conjugates were synthesized by the solid phase method using standard Boc chemistry [15], after which the PNA moiety was extended from the N-terminus of the peptide by manual Boc coupling according to Christensen et al [14] To introduce the

Correspondence to J Oehlke, Institute of Molecular Pharmacology,

Robert-Ro¨ssle-Str 10, D-13125 Berlin, Germany.

Fax: + 49 30 94793 159, Tel.: + 49 30 94793 267,

E-mail: oehlke@fmp-berlin.des

Abbreviations: CE-LIF, capillary electrophoresis with laser-induced

fluorescence detection; CLSM, confocal laser scanning microscopy;

CM, spontaneously-beating neonatal rat cardiomyocytes; CPP,

cell-penetrating peptide; DPBSG, Dulbecco’s phosphate buffered

saline/glucose; FACS, fluorescence-activated cell sorting;

Fluos, 5(6)-carboxyfluorescein-N-hydroxysuccinimide ester; MAP,

model amphipathic peptide; PNA, peptide nucleic acid.

(Received 16 March 2004, revised 21 May 2004,

accepted 28 May 2004)

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fluorescent label, the unprotected N-termini of the PNAs or

the PNA–MAP conjugates were reacted in

dimethylform-amide for 3 days at room temperature with 10 equivalents

of 5(6)-carboxyfluorescein-N-hydroxysuccinimide ester

(Fluos; Boehringer, Mannheim, Germany) Purification

was carried out by semipreparative HPLC on Vydac C18

using a 250· 8 mm column MALDI-MS (Voyager-DE

STR BioSpectrometry Workstation MALDI-TOF;

Persep-tive Biosystems, Inc., Framingham, MA, USA) provided

the expected [M + H]+ peaks (3878 and 5609 Da for I

and II, III, respectively)

Cell culture

CHO cells were cultured in 24-well plates (5· 104cells per

well) or for CLSM on 22· 22 mm coverslips (2 · 104cells)

at 37C in a humidified air environment containing 5%

CO2 in Ham’s F-12 medium supplemented with

290 mgÆmL)1glutamine and 10% (w/v) fetal bovine serum

Spontaneously-beating, neonatal rat cardiomyocytes (CM)

were obtained from ventricles of 1–2 day-old Sprague–

Dawley rats and cultured as described earlier [16]

Experi-ments conformed with the Guide for the Care and Use of

Laboratory Animals (NIH) and were approved by the local

government

The chronotropic response of the CM was measured as

described previously [16] on day 4 after seeding, every 5 min

after cumulative addition of nociceptin/orphanin FQ

(FGGFTGARKSARKLANQ) [17,18] at 37C Antisense

pretreatment of the heart cells was performed on days 1 and

2 after seeding by administration of either 0.2 lMPNA or

PNA–MAP conjugate

Assessment of cellular uptake by CLSM

The CLSM measurements were performed using a LSM

410 inverted confocal laser scanning microscope (Carl Zeiss,

Jena GmbH, Jena, Germany) as described previously

[19, 20] In brief, the fluorescent oligonucleotide derivatives

were dissolved in 1 mL prewarmed (37C) Dulbecco’s

phosphate buffered saline supplemented with 1 gÆL)1

D-glucose (DPBSG) and the cells were overlaid with this

solution within 5 min After 30 min observation, the

viability of the cells was assessed by the addition of trypan

blue Excitation was performed at 488 nm (Fluos) and

543 nm (trypan blue) and emission was measured at 515 nm

and 570 nm, respectively Three regions of interest (16· 16

pixel; 30 scans with a scan time of 2 s with double

averaging) in the cytosol and one in the nucleus of three

selected cells were chosen such that the intensity of the

diffuse fluorescence could be recorded without substantial interference from vesicular fluorescence The intracellular fluorescence signal was corrected for the contribution of the extracellular fluorescence, arising from nonideal confocal properties of the CLSM, by estimating the distribution function of sensitivity in the z direction of the microscope Assessment of cellular uptake by FACS

The cells (105 per well) were washed three times with prewarmed DPBSG and then overlaid with 0.2 mL of a freshly prepared prewarmed (37C) solution of the fluor-escent PNA derivative in DPBSG (2 lM I or 0.1 lM II; Table 1) After 30 min incubation at 37C, the cells were washed two times with NaCl/Piand detached by 15 min trypsination at 37C using 0.5 mL 0.05% Trypsin/0.02% EDTA (v/v) per well Then 1 mL culture medium was added and the cell suspension was centrifuged at 1000 g for

8 min Subsequently the cells were resuspended in 1 mL NaCl/Pi and stored on ice until the measurement The accumulation of fluorescence was determined at 525 nm after excitation at 488 nm using a Becton Dickinson (Franklin Lakes, NJ, USA) FACS Calibur flow cytometer with CELLQUESTsoftware Cytograms were acquired with

104cells

Assessment of cellular uptake by CE-LIF The cells were overlaid with 0.2 mL of a prewarmed (37C) solution of the fluorescent oligonucleotide derivative in DPBSG (0.5 lM I; 0.2 lM II; Table 1) immediately after addition of the respective aliquot of the sonicated PNA stock solution to the DPBSG After 30 min incubation at

37C (if not indicated otherwise), the cells were washed four times with ice-cold NaCl/Piand lysed for 2 h at 0C with 0.2 mL 0.1% (v/v) Triton X-100 containing 10 mmolÆL)1 trifluoroacetic acid The lysate, which contained only negligible amounts of fluorescent PNA derivatives (below 10% of total cell-associated PNA) was used for protein determination according to the method of Bradford [21] The wells containing attached cell debris and nuclei along with bound or precipitated PNA derivative were extracted

by sonication for 5 min at 60C with 0.2 mLÆwell)1Tris/ borate buffer (20 mM, pH 7.5) supplemented with 5Murea, 0.1% (w/v) SDS and, as an internal standard, 10 nM e-fluoresceinyl lysine The resulting extracts were centri-fuged for 3 min at 3000 g and stored at)20 C; immedi-ately prior to the CE-LIF analysis the extracts were sonicated for 5 min at 60C

CE-LIF was performed using a P/ACE MDQ system with a P/ACE MDQ Laser-Induced Fluorescence Detector (Beckman Coulter, Fullerton, CA, USA) and a

CZESep-600 neutral coated capillary (31 cm, 50 lM i.d.; Pheno-menex, Aschaffenburg, Germany) Tris/borate (200 mM,

pH 7.5) with 5Murea and 0.1% (w/v) SDS was used as the running buffer The cell extracts were injected into the capillary for 5 s at 0.5 p.s.i and the separations were performed at 650 VÆcm)1 and 25C The peaks of the references appeared after 1.8 min (I), 2.1 min (e-fluorescei-nyl lysine) and 4.2 min (II) Apart from free I and II, the cell extracts contained the largest quantity of compound I or II

in a complex bound form appearing in both cases at

Table 1 Sequences of the PNA derivatives studied.

Compound Sequence

MAP KLALKLALKALKAALKLA-NH 2

(antisense)

(antisense) III Fluos-AGGAGCAGGGAA-MAP

(scrambled)

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3.9 min The assignment for this peak is confirmed by its

complete disappearance after addition of an excess of 2 lM

unlabelled I and the concomitant generation of equally

intensive fluorescent peaks at the positions of the pure

reference compounds I and II, respectively

Quantitation was performed by fluorescence

measure-ment at 520 nm after excitation at 488 nm using an argon

ion laser The peaks were integrated using theP/ACE-SYSTEM

MDQ software (Beckman Coulter, Fullerton, CA, USA),

and were normalized to the area of the internal standard

e-fluoresceinyl lysine in order to eliminate irregularities of

injection and buffer status Because the exact volume of the

sample injected into the capillary remained unknown, the

references used as calibration standards were injected under

essentially the same conditions in order to eliminate this

factor in the subsequent calculations The concentrations of

the references were determined on the basis of the optical

density at 260 nm and proved linearly correlated to the peak

areas in the range between the quantitation limits and

500 nM The quantitation limits (signal-to-noise ratio > 3)

were about 0.5 pmolÆmL)1and 1.5 pmolÆmL)1for I and II,

respectively

Results

Conjugation with MAP leads to an increased intracellular

availability of PNA

In order to examine the ability of MAP to deliver PNA into

intact cells, we investigated the cellular uptake of the

conjugate of I with MAP in comparison with that of naked

I, by means of FACS, CLSM and CE-LIF The former two

protocols have been most widely used so far in such context

The results obtained in this way, however, were suspected

recently to be biased by surface adsorption or fixation

artefacts [22, 23] Moreover, due to the environmental

dependence of the fluorescence intensity, these approaches

only enable relative quantitative conclusions Therefore, we

have developed the third, a CE-LIF based protocol, which

appears capable of supplementing FACS and CLSM by

providing absolute quantitation of internalized PNA

CLSM revealed extensive fluorescence in the cytosol and

nucleus of CM and CHO cells after exposure to both the

naked PNA I and its MAP-conjugate (compound II)

(Fig 1) The intensity of this fluorescence (outside of

vesicles) in all cases was of the same order as that of the

extracellular PNA solution, indicating extensive

nonendo-cytic uptake for both I and II No differentiation between

the permeation behavior of I and II appears possible on the

basis of the CLSM data, except that a lower rate of

re-export became apparent for the conjugate (Fig 1)

FACS, on the other hand, revealed clearly higher

cell-associated fluorescence even after exposure to 100 nM

conjugate, than found after incubation with 2 lM of the

naked PNA (Fig 2) However, in this case, surface

adsorption of the conjugate, combined with washout of

the naked PNA due to the peptide tag and the relatively

long time required for the wash and trypsination processes,

respectively, might have biased the results

In addition to the information provided by CLSM and

FACS, the results obtained by the CE-LIF approach

enab-led a quantitative differentiation between the intracellular

concentrations of I and II If related to the external concentrations, the intracellular concentration of the con-jugate measured by CE-LIF in CM exceeded that of the naked PNA by about eightfold (Fig 3), which correlates well with the respective bioactivities (see below) Complementary uptake experiments performed with more conveniently available CHO cells analogously revealed a significantly higher uptake of the conjugate (Fig 3) Washout effects should influence the CE-LIF results only

to a negligible extent, considering that 3 min at 0C and

15 min at 37C are required for the wash process and for the efflux of about 50% of the internalized naked PNA (Fig 1), respectively That surface adsorption should also

Fig 1 Fluorescence intensity measured by CLSM according to [19] in cytosol and nucleus of CHO cells and cardiomyocytes Exposure to 0.5 l M I or 0.2 l M II for 30 min at 37 C and subsequent re-exchange (RE) into empty buffer for 15 min at 37 C, normalized to the fluor-escence intensity of the external PNA solution Each bar represents the mean of three samples ± SEM.

Fig 2 Cell-associated fluorescence measured by flow cytometry after exposure of CHO cells for 30 min at 37 °C to empty buffer, com-pound I and comcom-pound II Empty buffer (thin line; mean fluorescence intensity 2.6), to 2 l M I (broken line; mean fluorescence intensity 5.7)

or to 0.1 l M II (bold line; mean fluorescence intensity 7.3) Cell number

is plotted on the ordinate as a function of the fluorescence intensity on the abscissa.

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not interfere decisively with the quantification of the

internalized PNA by CE-LIF is implied by a comparison

of the uptake results depicted in Fig 3 As the extent of

surface adsorption should be comparable in all cases, its

contribution to the uptake results should be confined to the

lowest values found, which were assessed for the uptake into

CHO cells at 0C (Fig 3) Thus, the bias by surface

adsorption of the other results should amount at maximum

to about 20% and 50% for the naked PNA and the

conjugate, respectively The real bias, however, should

clearly be lower than these values because the values found

at 0C, at least in part, should also reflect

energy-independent uptake Consistent with these notions in the

extracts of cells exposed to an analogous disulfide bridged

PNA–MAP-conjugate (Y Wolf, M Bienert & J Oehlke,

unpublished observation) no surface bound conjugate

above the quantification limit of CE-LIF could be detected

In this case exclusively the naked PNA generated by

cleavage of the disulfide bond in the reducing environment

of the cell interior was found

Energy-dependent and energy-independent mechanisms

are involved in the cellular uptake of both naked PNA

and its MAP conjugate

The cellular uptake of both the naked PNA and the

conjugate proved only partially sensitive to lowered

temperature and energy depletion, implying the

involve-ment of nonendocytic mechanisms (Fig 3) On the other

hand, energy-dependent and -independent mechanisms

contributed differently to the cellular uptake of the naked

PNA and of the conjugate (Fig 3), suggesting that distinct

modes were functioning in the two cases The different

sensitivity to the presence of serum observed for the

internalization of the naked PNA and its conjugate,

respectively, probably also suggests distinct modes of uptake (Fig 3), although different association with serum components appear more likely to be the reason here

Intracellular PNA concentration increases more rapidly after exposure of cells to PNA–MAP-conjugate than to naked PNA

The quantity of cell-associated PNA increased significantly faster after exposing CHO cells to II than after incubation with I ( Figs 4 and 5) This finding unravels a further reason for the enhanced bioavailability of the PNA–MAP conju-gate, besides reduced re-export and resistance to serum influences mentioned above After 60 min the cell-associ-ated concentration of the conjugate reached a level at which apparently the efflux equalled the influx, whereas that of the naked PNA increased linearly further (Fig 4) Possible alternative reasons for the observed arrest of the uptake of

Fig 3 Amount of cell-associated PNA determined by CE-LIF in the

extracts of CHO cells and cardiomyocytes Results following exposure

to 0.5 l M I or 0.2 l M II for 30 min at 37 C without (control) and with

energy depletion or in the presence of 10% (v/v) fetal bovine serum

(FBS) and at 0 C For energy depletion, the cells were incubated in

DPBS containing 25 m M 2-deoxyglucose/10 m M sodium azide (DOG/

NaN3) for 60 min at 37 C and subsequently exposed to the PNA

derivative dissolved in the same buffer To facilitate comparison, the

values of I were normalized to an exposure at 0.2 l M according to the

linear concentration dependence shown in Fig 7 Each bar represents

the mean of three samples ± SEM.

Fig 4 Cell-associated PNA determined by CE-LIF in the extracts of CHO cells after exposure to 0.5 l M I (r) or 0.2 l M II (j), respectively,

at 37 °C for different periods of time To facilitate comparison, the values of I were normalized to an exposure at 0.2 l M according to the linear concentration dependence shown in Fig 7 Each bar represents the mean of three samples ± SEM.

Fig 5 Cell-associated PNA determined by CE-LIF in the extracts of CHO cells after exposure to various concentrations of I (r) or II (j) for

30 min at 37 °C Each bar represents the mean of three samples

± SEM.

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II after 60 min could be aggregation of the conjugate and

peptidase cleavage of the MAP-tag However, even after 2 h

exposure to the cells no significant loss of II was detectable

by CE-LIF in the supernatants, ruling out aggregation and

enzymatic breakdown from playing a noticeable role in this

context

Conjugation with MAP significantly augments the

biological activity of a 12-mer antisense PNA directed

against the nociceptin/orphanin FQ receptor

Pretreatment of CM with 0.2 lM of the naked PNA

(I; Table 1) and its MAP-conjugate (II; Table 1) lowered

the chronotropic effect exerted by the neuropeptide

noci-ceptin [24] by 50% and 90%, respectively ( Figs 6 and 7)

Exposure of CM to conjugates of MAP with a scrambled

PNA containing the same base composition

(com-pound III; Table 1) did not negatively influence the

chronotropic effect (Fig 7) These results infer, as anticipa-ted, antisense down-regulation of the nociceptin/orphanin

FQ receptor to be the mechanism of biological activity of compounds I and II The antisense pretreatment remained without any influence on the basal beating rate of the CM, implying that the PNA derivatives are nontoxic at the concentration used Consistent with this notion, no signi-ficant signs of toxicity were found by means of LDH release and trypan blue exclusion throughout all cellular uptake experiments of the present study

Discussion Improved delivery into mammalian cells has been achieved

by covalent coupling of various highly polar bioactive substances with CPPs [10,25–27] The structural require-ments for the delivery activity of peptides, however, remained unclear until now As a prerequisite for an elucidation of such structural requirements in the present work we evaluated the suitability of a conjugate of the synthetic CPP MAP

(KLALKLALKALKAALKLA-NH2) [9,10] with a 12-mer peptide nucleic acid (5¢-GGAGCAGGAAAG-3¢) directed against the mRNA

of the nociceptin/orphanin FQ receptor [12,13] to serve as the parent compound in planned structure–delivery activity relationship investigations

In distinction to earlier studied conjugates of MAP with phosphorothioate oligonucleotides [11] the MAP–PNA conjugates proved nontoxic in the concentration range up

to 1 lM Consistent with previous reports about substan-tially enhanced bioavailability of PNA after conjugation with natural CPPs [3–8], an almost one order of magnitude higher intracellular PNA concentration was achieved after exposing cells to the MAP–PNA conjugate II than after incubation with the naked PNA Correspondingly, pretreat-ment of CM with II impaired the chronotropic effect elicited

by the neuropeptide nociceptin [16–18,24] more than five-fold more than preincubation with the naked PNA

In line with a nonendocytic mode of uptake regarded to

be typical of CPPs [10,25,27], a faint cytosolic and nuclear fluorescence of comparable intensity than measured exter-nally was detected by CLSM in cells treated with II in addition to a clearly visible vesicular fluorescence Surpris-ingly, however, a similar pattern was observed after exposing the cells to the naked PNA, suggesting nonend-ocytic as well as endnonend-ocytic uptake mechanisms to be involved in both cases Quantitation of the intracellular PNA by CE-LIF supported this notion The measured quantities corresponded to more than 20-fold and fourfold enrichments in the cell interior for the MAP-conjugate and the naked PNA, respectively (related to the external concentration and a ratio of about 10 lL cell volumeÆmg protein)1[11]) If the internalization had proceeded primar-ily by an endocytic mechanism, an intracellular concentra-tion of, at maximum, about 10% of the external level should

be expected, considering the total volume of endosomes comprising maximally 10% of the cell volume [28] Thus the commonly accepted interpretation of the different cellular uptake of naked PNA and PNA–CPP conjugates by an endocytic internalization of the former and a nonendocytic one of the latter appears insufficient for explaining our results As one of the possible explanations, our observation

Fig 6 Dose–response curve for the influence of nociception on the

beating rate of CM measured on day 4 of culture without and with

pretreatment with either 0.2 l M of I or II on day 1 and 2 after seeding.

The basal beating rate of the CM on day 4 of culture was 191 ± 12

beatsÆmin)1(SD; n¼ 30) Each point represents the mean of 30–40

cells or cell cluster ± SEM.

Fig 7 Influence of 10)5M nociception on the beating rate of CM

measured on day 4 of culture without and with pretreatment with either

0.2 l M of II or III (Table 1) on day 1 and 2 after seeding Each point

represents the mean of 30–40 cells or cell cluster ± SEM The

differ-ence between the control and the asterisk-marked bar is statistically

significant at P £ 0.05 (Student’s t-test).

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that energy-dependent and -independent mechanisms

con-tribute differently to the cellular uptake of the naked PNA

and of the conjugate suggests that distinct modes of

nonendocytic uptake should be operative in both cases

Our findings that the MAP–PNA conjugate in contrast to

the naked PNA proved resistant to re-export provide a

further explanation of the different intracellular levels

measured for the conjugate and the naked PNA Analogous

different susceptibilities against re-export were observed for

phosphorothioate oligonucleotides and

MAP–phosphoro-thioate oligonucleotide conjugates [11], suggesting that such

peptide mediated effects are not confined to particular cargo

molecules In addition, the uptake differences between

conjugate and naked PNA were strengthened by serum

influences Similarly Astriab-Fischer et al [29] found that

the presence of serum in the incubation medium did not

affect the uptake of CPP–phosphorothioate oligonucleotide

conjugates in contrast to that of the naked oligonucleotides,

infering that such peptide mediated resistance to serum

influences is a more general reason for the improved

bioavailability of CPP conjugates

In conclusion, our study revealed the ability of the

synthetic CPP MAP to significantly increase the

bioavail-ability and bioactivity of PNA without eliciting enhanced

cell toxicity These results along with the possibility to

measure absolute quantities of internalized PNA by

CE-LIF provide promising preconditions for studies on

struc-ture–delivery activity relationships which are under way

Acknowledgements

We thank H Hans, M Wegener and G Vogelreiter for excellent

technical assistance This work was supported by the European

Commission (QLK3-CT-2002-01989).

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