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Here we report identification of growth factor-regulated miRNAs using the PC12 cell line, an established model of neuronal growth and differentiation.. We found that expression of miR-221

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microRNA-221 and 222 in PC12 cells

Kazuya Terasawa1, Atsuhiko Ichimura1, Fumiaki Sato2, Kazuharu Shimizu2and Gozoh Tsujimoto1

1 Department of Pharmcogenomics, Graduate School of Pharmaceutical Sciences, Kyoto University, Sakyo-ku, Japan

2 Department of Nanobio Drug Discovery, Graduate School of Pharmaceutical Sciences, Kyoto University, Sakyo-ku, Japan

MicroRNAs (miRNAs) are evolutionally conserved

small non-coding RNAs that regulate gene

expres-sion at the post-transcriptional level and play

impor-tant roles in a wide variety of biological functions,

including cell differentiation, tumorigenesis, apoptosis

and metabolism [1–3] Approximately 30% of human

protein-coding genes are predicted to be targets of

miRNA [4,5] Biogenesis of miRNA and the

mecha-nism for regulation of target gene expression by

miRNA are relatively well characterized miRNA

genes are initially transcribed mainly by RNA

poly-merase II as long primary transcripts (pri-miRNAs),

processed by the nuclear RNase Drosha to produce

precursor miRNAs (pre-miRNAs), and then exported

to the cytoplasm Pre-miRNAs are further processed

into mature miRNAs by the cytoplasmic RNase

Dicer [6] miRNAs recognize and bind to partially complementary sites in the 3¢ UTRs of target mRNAs, resulting in either translational repression

or target degradation [7] To further understand the functional significance of miRNA, the regulatory mechanism of miRNA expression needs to be better understood

The mitogen-activated protein kinase (MAPK) cascades play an essential role in transducing extra-cellular signals to cytoplasmic and nuclear effectors, and regulate a wide variety of cellular functions, including cell proliferation, differentiation and stress responses [8,9] Extracellular signal-regulated kinases

1 and 2 (ERK1⁄ 2) were the first recognized members

of the MAPK family of proteins These kinases are primarily activated by mitogenic factors, differentiation

Keywords

Bim; ERK1 ⁄ 2; microRNA; NGF; PC12

Correspondence

G Tsujimoto, 45-29

Yoshida-Shimo-Adachi-cho, Sakyo-ku, Kyoto 606-8501, Japan

Fax: +81 75 753 4523

Tel: +81 75 753 4544

E-mail: gtsuji@pharm.kyoto-u.ac.jp

(Received 18 December 2008, revised 13

March 2009, accepted 6 April 2009)

doi:10.1111/j.1742-4658.2009.07041.x

MicroRNAs (miRNAs) are small non-coding RNAs that regulate gene expression by inhibiting translation and⁄ or inducing degradation of target mRNAs, and they play important roles in a wide variety of biological func-tions including cell differentiation, tumorigenesis, apoptosis and metabo-lism However, there is a paucity of information concerning the regulatory mechanism of miRNA expression Here we report identification of growth factor-regulated miRNAs using the PC12 cell line, an established model of neuronal growth and differentiation We found that expression of miR-221 and miR-222 expression were induced by nerve growth factor (NGF) stim-ulation in PC12 cells, and that this induction was dependent on sustained activation of the extracellular signal-regulated kinase 1 and 2 (ERK1⁄ 2) pathway Using a target prediction program, we also identified a pro-apo-totic factor, the BH3-only protein Bim, as a potential target of

miR-221⁄ 222 Overexpression of miR-221 or miR-222 suppressed the activity of

a luciferase reporter activity fused to the 3¢ UTR of Bim mRNA Further-more, overexpression of miR-221⁄ 222 decreased endogenous Bim mRNA expression These results reveal that the ERK signal regulates miR-221⁄ 222 expression, and that these miRNAs might contribute to NGF-dependent cell survival in PC12 cells

Abbreviations

EGF, epidermal growth factor; ERK, extracellular signal-regulated kinase; MAPK, mitogen-activated protein kinase; MEK, MAPK/ERK kinase; miRNA, microRNA; NGF, nerve growth factor.

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stimuli and cytokines such as nerve growth factor

(NGF) and epidermal growth factor (EGF) [10–12]

Because both ERK signaling and miRNA function are

involved in a variety of important biological responses,

the significance of ERK signaling in terms of regulating

miRNA expression is of great interest

To study the role of the ERK1⁄ 2 pathway in the

regulation of miRNA expression, we first determined

the expression profile of miRNAs by using the

growth factor-induced neural differentiation process

of PC12 as a model It is well known that NGF,

but not EGF, induces neural differentiation in PC12

cells, although both growth factors potently activate

ERK1⁄ 2 [13–16] We identified miR221 and 222 as

differentially regulated miRNAs Although the

expression of these miRNAs was found to be

ERK-dependent, the effect of NGF and EGF on their

expression was different; thus, only NGF, but not

EGF, can induce their expression Further, our study

showed that the sustained activation of ERK1⁄ 2 by

NGF, but not the transient activation of ERK,

could effectively induce miR221 and 222 in PC12

cells Finally we identified the BH3-only protein

Bim, which is involved in NGF-dependent neuronal

survival [17–19], as a potential target of miR-221

and 222

Results

NGF stimulation induces miR-221/222 in PC12 cells

Treatment of PC12 cells with NGF for 48 h induced readily detectable neurite outgrowth (Fig 1A), so we selected two points at 0 and 48 h after stimulation to compare the expression of miRNAs We used a TaqMan miRNA assay, featuring reverse transcription using stem-loop RT-PCR primers followed by real-time PCR using TaqMan probes [20], to examine the expression of

156 rat miRNAs We identified only two miRNAs, miR-221 and 222, as drastically up-regulated 48 h after NGF stimulation (data not shown) These miRNAs are encoded in tandem on the chromosome X (Fig 1B) Next, we examined the time course of expression of miR-221 and 222 Quantitative RT-PCR analysis showed that these miRNAs had a very similar profile (Fig 1C) An alternative RT-PCR analysis, using a set

of primers that amplify a fragment located between these two miRNAs, demonstrated transcriptional induction of this region upon NGF stimulation (data not shown) These data support the notion that

miR-221 and 222 derive from the same pri-miRNA [21] Following NGF stimulation, the expression level of

Fig 1 NGF induces expression of the microRNAs miR-221 and 222 in PC12 cells (A) NGF-induced differentiation of PC12 cells (B) Schematic representation of the genomic structure of miR-221 and 222 and their corresponding sequences (C, F) PC12 cells were treated with 100 ngÆmL)1NGF or

30 n M EGF for the indicated times Cells were harvested and total RNA was pre-pared The RNA was subjected to quantita-tive RT-PCR to assess the levels of mature miR-221 ⁄ 222 The data represent means

of the Ctvalues (± SD, n = 3) In (C),

*P < 0.01 for miR-221 versus 0 h point, and



P < 0.01 for miR-222 versus 0 h point In (F), P < 0.01 for comparisons indicated by asterisks (Tukey’s test) (D, E) Cell extracts were subjected to immunoblotting with a-phospho-ERK1 ⁄ 2 and a-ERK1 ⁄ 2 IgGs.

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both miRNAs rapidly increased and reached a

maximum at 3–6 h, which was then sustained to the

last time point assayed at 48 h Over this time course,

expression of an internal control U6 remained

unchanged (Fig 1C) Based on the threshold cycle (Ct)

changes, we estimate that the expression of miR-221

and 222 had increased by approximately 26-fold NGF

induced sustained activation of ERK1⁄ 2 over this time

course (Fig 1D) We further examined whether EGF

stimulation also induces miR-221⁄ 222 in PC12 cells

Our analysis confirmed that EGF transiently activated

ERK1⁄ 2, unlike NGF [13–16] (Fig 1E) Because

NGF-mediated expression of miR-221⁄ 222 peaked

approximately 3 h after stimulation, we examined the

expression of miR-221⁄ 222 at this time point in all

further experiments In contrast to NGF stimulation,

EGF did not induce any detectable up-regulation of

miR-221 and 222 at 3 h (Fig 1F) Moreover, no such

stimulation was found even after monitoring for up to

48 h (data not shown)

Sustained activation of ERK1/2 is necessary for

induction of miR-221/222

We next studied whether the NGF-induced expression

of miR-221⁄ 222 depends on ERK1 ⁄ 2 activation We

first examined the effect of a specific inhibitor (U0126)

for MAPK⁄ ERK kinase (MEK) 1 ⁄ 2, which is a direct

activator of ERK1⁄ 2 [22] As shown in Fig 2A,

pre-treatment of U0126 potently inhibited NGF-induced

ERK1⁄ 2 activation The same pre-treatment with

U0126 completely blocked induction of miR-221 and

222 (Fig 2B) Moreover, we found that expression of

miR-221⁄ 222 dramatically increased when

constitu-tively active MEK1 (MEK1SDSE) [23] was transiently

expressed in PC12 cells (Fig 2C,D)

Taken together, these results indicate that induction

of miR-221⁄ 222 depends on the activation of ERK1 ⁄ 2

However, transient activation of ERK⁄ 2 upon EGF

stimulation did not induce miR-221⁄ 222 expression

This observation prompted us to hypothesize that

induction of these miRNAs requires sustained

activa-tion of ERK1⁄ 2 To verify this hypothesis, we blocked

NGF-induced sustained ERK1⁄ 2 activation by

add-ing U0126 10 min after NGF treatment (Fig 3A) As

shown in Fig 3B, addition of U0126 completely

inhib-ited the sustained activation of ERK1⁄ 2 In this

situa-tion, the induction of miR221⁄ 222 was also completely

suppressed (Fig 3C) These results clearly demonstrate

that sustained activation of ERK1⁄ 2 is required for

induction of miR-221 and 222

However, the apparent induction of these miRNA

molecules was only observed approximately 3 h

after NGF stimulation (Fig 1C), which implies that miR-221 and 222 are not the primary target genes regulated by NGF We reasoned that this induction requires de novo protein synthesis As shown in Fig 4A, treatment with cycloheximide completely inhibited induction of these miRNAs, but had no significant effect on ERK1⁄ 2 activation (Fig 4B) We also confirmed inhibition of protein synthesis by monitoring expression of c-Fos protein, which is a

Fig 2 Activation of ERK1 ⁄ 2 pathway is involved in miR-221 ⁄ 222 induction (A, B) PC12 cells were pre-treated with 10 l M U0126 for

10 min before treatment with 100 ngÆmL)1NGF for 3 h Inhibition

of ERK1 ⁄ 2 activity by U0126 was confirmed by immunoblotting (A) The expression levels of miR-221 ⁄ 222 were examined as described

in Fig 1C (*P < 0.05, Tukey’s test) (B) (C, D) PC12 cells were transfected either with empty vector or HA-tagged MEK1SDSE The expression levels of miR-221 ⁄ 222 were examined 24 h after transfection as described in Fig 1C (*P < 0.05 versus empty vec-tor, Student’s t test) (C) Expression of HA-tagged MEK1SDSE was confirmed by immunoblotting (D) DMSO, dimethylsulfoxide.

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well-known NGF-induced immediate early gene [24].

Cycloheximide treatment completely blocked c-Fos

protein synthesis (Fig 4C, compare with control)

These data indicate that de novo protein synthesis is

required for the induction of miR-221⁄ 222

Pro-apototic Bim is a plausible target of

miR-221/222

We used TargetScan [4] to identify the likely target

genes of miR-221⁄ 222 Specifically, we focused on

the pro-apototic Bim gene because Bim has been

reported to be involved in NGF-dependent survival

of PC12 cells [19] The predicted target site for these

miRNAs is conserved in human, mouse, rat, dog

and chicken (Fig 5A) The rat Bim gene had no

annotated 3¢ UTR, and so in the TargetScan

pro-gram this is computationally determined based on

the human Bim 3¢ UTR sequence Initially, we used

RACE to verify whether the predicted 3¢ UTR

region is transcribed 3¢ RACE analysis detected

products containing the terminal portion of the pre-dicted Bim 3¢ UTR Moreover, RT-PCR analysis showed that a fragment containing the predicted tar-get site was amplified (data not shown) To examine whether these miRNAs can target the Bim gene, we generated a luciferase construct harboring a fragment

of the Bim 3¢ UTR containing the target sequence (Fig 5A) Co-expression of either 221 or

miR-222 significantly (P < 0.05) suppressed the reporter activity compared to the control (Fig 5C, wt) Muta-tions introduced into the predicted binding site almost eliminated this suppression These results sug-gest a direct interaction of these miRNAs with the predicted target site of the Bim 3¢ UTR (Fig 5B,C, mt) Furthermore, we investigated the effect of expres-sion of miR-221⁄ 222 on endogenous Bim mRNA expression Because the Bim gene has three alternative splice variants [25], we designed a set of primers to detect all three products Overexpression of either miR-221 or miR-222 resulted in significant (P < 0.05)

A

B

C

Fig 3 Sustained activation of ERK1 ⁄ 2 is required for NGF-induced

miR-221 ⁄ 222 expression (A) Schematic diagram of the

experimen-tal design (B, C) PC12 cells were pre-treated with 10 l M U0126

for 10 min after treatment with 100 ngÆmL)1NGF for 3 h Inhibition

of sustained ERK1 ⁄ 2 activation by U0126 was confirmed by

immu-noblotting (B) The expression levels of miR-221 ⁄ 222 were

exam-ined as described in Fig 1C (*P < 0.05, Tukey’s test) (C) DMSO,

dimethylsulfoxide.

Fig 4 Protein synthesis is required for NGF-induced miR-221 ⁄ 222 expression PC12 cells were pre-treated with 10 lgÆmL)1 cyclohexi-mide for 30 min before treatment with 100 ngÆmL)1NGF for 3 h The expression levels of miR-221 ⁄ 222 were examined as described

in Fig 1C (*P < 0.05, Tukey’s test) (A) ERK1 ⁄ 2 activation in the presence of cycloheximide (B) and inhibition of protein synthesis by cycloheximide (C) were confirmed by immunoblotting a-Tubulin was used as a loading control DMSO, dimethylsulfoxide.

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down-regulation of Bim mRNA (Fig 5D) To show that

this down-regulation is a specific effect for Bim mRNA,

we examined the mRNA level of an apoptosis-related

gene, Bax, because the 3¢ UTR of Bax mRNA has no

predicted target site for miR-221⁄ 222 We found that

overexpression of either miR-221 or miR-222 had no

significant effect on the Bax mRNA level (data not shown) Taken together, our data suggest that miR-221 and 222 can target the Bim gene

Discussion

The present study has demonstrated that, in PC12 cells, miR-221 and 222 are transcriptionaly induced upon stimulation by NGF, and that this induction requires sustained ERK1⁄ 2 activation and de novo pro-tein synthesis Presumably, sustained ERK1⁄ 2 activa-tion is required for the inducactiva-tion of transcripactiva-tional regulatory protein(s) that regulates miR-221⁄ 222 expression Recently, miR-155 expression has been shown to be regulated by two MAPK pathways, the ERK1⁄ 2 and c-Jun N-terminal kinase pathways, through AP-1 signaling [26] AP-1 family proteins are good candidates for mediating NGF-induced miR-221⁄ 222 expression in PC12 cells

A previous study showed that miR-221 and 222 are up-regulated in quiescent cells that have been stimulated to proliferate by serum stimulation [27] ERK1⁄ 2 is known to play a critical role in growth factor-stimulated cell-cycle progression from G0⁄ G1

to S phase, and sustained activation is required for this progression [28] Our finding that sustained ERK1⁄ 2 activation induces miR-221 ⁄ 222 expression

is entirely consistent with this observation miR-221

Fig 5 The Bim 3¢ UTR is a target of miR-221 ⁄ 222 (A) Schematic representation of the reporter construct and conservation of the tar-get site of miR-221 ⁄ 222 in the Bim 3¢ UTR in vertebrates The boxed region indicates the sites complementary to the seed sequence of miR-221 ⁄ 222 (B) Predicted base pairing between miR-221 and 222 and their target sites in the Bim 3¢ UTR Underlin-ing indicate the seed sequences The calculated free energy of hybridization of each miRNA with the target site is also indicated These data were prepared using RNAhybrid Lower-case letters indicate the sites introduced to the Bim 3¢ UTR by mutation The indicated bases were substituted with complementary nucleotides

in the mutation construct (C) The indicated RNA oligonucleotides (10 pmol per well) and reporter plasmid (200 ng per well) were co-transfected with the internal control plasmid (20 ng per well) into PC12 cells After 24 h, the cells were harvested, and the lysates were subjected to a luciferase assay The results are the ratio of firefly to renilla luciferase activity (mean ± SD, n = 3) The lucifer-ase activity ratio for control RNA transfection (mock) for each repor-ter was set at 1 (*P < 0.01 versus mock; Student’s t test) (D) PC12 cells were transfected with the indicated RNA oligonucleo-tides After 24 h, the cells were harvested and total RNA was pre-pared The RNA was subjected to quantitative RT-PCR to assess the levels of Bim mRNA The Bim mRNA expression was normal-ized against GAPDH mRNA expression (mean ± SD, n = 3) The normalized Bim expression of control RNA transfection (mock) was set at 1 (*P < 0.05 versus mock; Student’s t test).

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and 222 have also been reported to be up-regulated

in some cancer cell lines and to function as

onco-genic miRNAs by targeting the cyclin-dependent

kinase inhibitor p27Kip1 [29–32] These studies

strongly suggest that miR-221 and 222 are involved

in the regulation of cell growth In PC12 cells, NGF

stimulation of starved cells promotes cell survival

and differentiation, and inhibits cell-cycle progression

[16] However, NGF-induced miR-221 and miR-222

expression in PC12 cells is probably not involved in

cell-cycle progression These apparently contradictory

effects might be attributed to cell type-dependent

differences

We could not observe any detectable effect of

NGF-induced neurite outgrowth when miR-221

and⁄ or 222 were overexpressed in PC12 cells (data

not shown) However, we show that the pro-apototic

protein Bim is a plausible target of miR-221⁄ 222

Bim is known to be regulated at both the

transcrip-tional and post-transcriptranscrip-tional level [33] Here, we

have confirmed that Bim is regulated at the

transla-tional level In PC12 cells, the Bim gene is induced

upon withdrawal of serum When NGF stimulation

occurs, activation of ERK1⁄ 2 causes phosphorylation

of Bim proteins and inhibits their function, resulting

in cell survival This translational regulation of Bim

ensures the down-regulation of Bim function and

hence cell survival These data indicate that

NGF-induced miR-221⁄ 222 expression is involved in

NGF-dependent cell survival Interestingly, recent reports

have shown that Bim is regulated by miR-32 and

the miR-17–92 cluster of miRNAs, which are

known to be up-regulated in several cancers [34–36]

Up-regulation of these miRNAs, which results in

down-regulation of pro-apoptotic Bim mRNA, exerts

an anti-apoptotic effect in some cancers [35,36] The

miR-17–92 cluster has also been shown to be

involved in B-cell development and to target Bim

mRNA [37,38] In PC12 cells, the expression level of

these miRNAs is moderate and remains unchanged

upon NGF stimulation (data not shown) Thus,

induced miR-221 and 222 might work cooperatively

with these miRNAs

miR-221 and 222 are highly conserved in

verte-brates and have the same seed sequence Moreover,

they are encoded in tandem on the same chromosome

in human, mouse and rat, indicating that they have

important roles in biological processes In this study,

we found that miR-221⁄ 222 expression is regulated

by the ERK1⁄ 2 pathway in PC12 cells This

regula-tion might also funcregula-tion in different biological

pro-cesses Our findings provide new insights into the

MAPK signaling pathway

Experimental procedures

Cell culture and transfection PC12 cells were maintained in Dulbecco’s modified Eagle’s medium plus 10% fetal bovine serum, 5% donor horse serum and antibiotics at 37C in 5% CO2 The cells were seeded onto poly-l-lysine-coated 60 mm dishes (AGC Techno Glass Co Ltd, Chiba, Japan) and incubated in a low concentration of serum (1% horse serum) for 24 h prior to treatment with 100 ngÆmL)1 NGF (Alomone Labs Ltd, Jerusalem, Israel) or 30 nm EGF (PeproTech EC Ltd, London, UK) Transfections were performed according to the manufacturer’s instructions using LipofectAMINE 2000 (Invitrogen, Carlsbad, CA, USA) The miRNA precursors miR-221 and 222 and control RNA were purchased from Ambion (Austin, TX, USA)

RNA isolation and RT-PCR analysis Total RNA was isolated using ISOGEN reagent (Nippon Gene Co Ltd, Tokyo, Japan) miRNA expression was mea-sured using TaqMan MicroRNA Assays (Applied Biosys-tems, Lincoln, CA, USA) according to the manufacturer’s protocol, except that all reactions were carried out at half scale The rat miRNAs assayed in this study are listed in the microrna assay index fileversion 1 (Applied Biosystems) U6 snRNA was used as an internal control For detection of Bim mRNA, reverse transcription was performed using a QuantiTect reverse transcription kit (Qiagen, Hilden, Ger-many) Prepared cDNAs were then subjected to quantitative PCR analysis using Power SYBR Green PCR Master Mix (Applied Biosystems) The primers for the PCR analysis were 5¢-CTTCCATAAGGCAGTCTCAG-3¢ and 5¢-CGGAAGA TGAATCGTAACAG-3¢ for Bim, and 5¢-TTGCTGACAA TCTTGAGGGAG-3¢ and 5¢-GAGTATGTCGTGGAGTC TACTG-3¢ for glyceraldehyde-3-phosphate dehydrogenase (GAPDH) The data were obtained and analyzed using an ABI 7300 real-time PCR system (Applied Biosystems)

Plasmid construction The plasmid pcDNA3-HA encoding Xenopus MEK1SDSE was supplied by E Nishida (Graduate School of Biostudies, Kyoto University, Japan) A partial fragment of the 3¢ UTR of rat Bim was amplified by PCR using the primers 5¢-CCTGCCTCTTGAGGTACTGC-3¢ and 5¢-AGCTAGT CGCAAGTTTTA-3¢ following reverse transcription from total RNA isolated from PC12 cells, and cloned into the pCR-Blunt vector (Invitrogen) Mutagenesis of the Bim 3¢ UTR was performed using a QuikChange site-directed mutagenesis kit (Stratagene, La Jolla, CA, USA) An EcoRI site was introduced into the XbaI site of the lucifer-ase reporter vector pGL4.23 (Promega, Madison, WI, USA) by ligation with the oligonucleotides 5¢-CTAGACT

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GAATTC-3¢ and 5¢-CTAGGAATTCAGT-3¢, yielding the

pGL4.23EcoRI vector EcoRI fragments of the wild-type

(wt) and mutated (mt) Bim 3¢ UTR forms of the

pCR-Blunt vector were ligated into the EcoRI site of the

pGL4.23EcoRI vector The identity of all constructs was

confirmed by DNA sequencing

Immunoblotting

Cells were harvested by scraping from culture dishes in hot

1· SDS sample buffer, and the lysates were separated by

SDS–PAGE and analyzed by immunoblotting Anti-HA

(3F10) rat monoclonal IgG was purchased from Roche

(Basel, Switzerland) Anti-p44⁄ 42 MAP kinase,

anti-phos-pho-p44⁄ 42 MAP kinase IgGs (numbers 9101 and 9102,

respectively) and anti-c-Fos IgG (number 4384) were

obtained from Cell Signaling (Danvers, MA, USA)

Anti-a-Tubulin (B-5-1-2) mouse monoclonal IgG was purchased

from Sigma (St Louis, MO, USA) Peroxidase-linked

sec-ondary antibodies were purchased from GE Healthcare

(Chalfont St Giles, UK) An LAS3000 CCD imaging

sys-tem (Fujifilm, Tokyo, Japan) was used for detection

Reporter assay

Cells grown in 24-well plates (1.0· 105cells per well) were

harvested for assays 24 h after transfection The luciferase

activity was measured using a dual-luciferase reporter assay

system (Promega) with a Lumat LB9507 luminometer

(Berthold Technologies, Bad Wildbad, Germany) As an

internal control, a renilla luciferase vector pGL4.70

(Pro-mega) was used The data represent means and standard

deviations of three independent experiments

Statistical analysis

The data were analyzed using Student’s t test or ANOVA

followed by Tukey’s test as indicated

Acknowledgements

This work was supported in part by a grant from the

Ministry of Education, Culture, Sports, Science and

Technology of Japan (to K.T., F.S., K.S and G.T.),

the New Energy and Industrial Technology

Develop-ment Organization (to K.T and G.T.), and the Uehara

Memorial Foundation (to G.T.)

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