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By performing cDNA microarray analyses of hypoxic hepa1c1c7 cells and BpRc1 cells, we identified both hypoxia-regulated genes and their Arnt dependence.. From the 259 genes, we selected h

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in an aryl hydrocarbon receptor nuclear translocator

(Arnt)-dependent manner

Su Mi Choi*, Hookeun Oh* and Hyunsung Park

Department of Life Science, University of Seoul, South Korea

Keywords

Arnt; gene expression; HIF; hypoxia;

microarray

Correspondence

H Park, Department of Life Science,

University of Seoul, 90 Jeonnong-dong,

Dongdaemun-gu, Seoul 130-743, South

Korea

Fax: +82 2 2210 2888

Tel: +82 2 2210 2622

E-mail: hspark@uos.ac.kr

*These authors made equal contributions to

this study

(Received 17 July 2008, revised

12 September 2008, accepted 17

September 2008)

doi:10.1111/j.1742-4658.2008.06686.x

We investigated hypoxia-inducible factor (HIF)-dependent changes in the expression of 5592 genes in response to hypoxia (0.1% O2, 16 h) by per-forming cDNA microarray analyses of mouse hepa1c1c7 and BpRc1 cells BpRc1 cells are a hepa1c1c7 variant defective in HIF-b⁄ aryl hydrocarbon receptor nuclear translocator (Arnt), and are therefore unable to induce HIF target genes in response to hypoxia By comparing hepa1c1c7 cells with BpRc1 cells, we were able to investigate hypoxia-regulated gene expression as well as the role played by HIF in regulating the hypoxic-dependent response of gene expression This study identified 50 hypoxia-induced genes and 36 hypoxia-repressed genes Quantitative PCR analysis

of nine genes confirmed our ability to accurately analyze changes in hypoxia-induced gene expression by microarray analysis By comparing quantitative PCR analyses of these nine genes in BpRc1 and hepa1c1c7 cells, we determined that eight of the nine hypoxia-induced genes are Arnt dependent Additional quantitative PCR analyses of eight hypoxia-repressed genes confirmed, with a 50% probability, that microarray analy-sis was able to predict hypoxia-repressed gene expression Only two of the four confirmed genes were found to be repressed in an Arnt-dependent manner Collectively, six of these 13 genes (46.2% probability) showed a pattern of expression consistent with the microarray analysis with regard to Arnt dependence Finally, we investigated the HIF-1a dependence of these

13 genes by quantitative PCR analysis in HIF-1a knockdown 3T3-L1 cells These analyses identified novel hypoxia-regulated genes and confirmed the role of Arnt and HIF-1a in regulating their expression These results identify additional HIF target genes and provide a more complete understanding of hypoxia signaling

Abbreviations

ABCC3, ATP-binding cassette, subfamily C (CFTR ⁄ MRP), member 3; Arnt, aryl hydrocarbon receptor (AhR) nuclear translocator; ATF-4, activating transcription factor-4; bHLH, basic helix–loop–helix; BNIP3, BCL-2 ⁄ adenovirus E1B 19 kDa-interacting protein 3; BSG, basigin; CCGN2, cyclin G2; DUSP12, dual specificity phosphatase 12; eIF1, eukaryotic translation initiation factor 1; ER, endoplasmic reticulum; FDR, false discovery rate; FKBP4, FK506 binding protein 4 (59 kDa); GNA11, guanine nucleotide binding protein, a 11; HIF, hypoxia-inducible factor; HSP60, heat shock protein, 60 kDa; IER3, immediate early response 3; MAD2L1, MAD2 (mitotic arrest deficient, homolog)-like 1 (yeast); MAPK, mitogen-activated protein kinase; MKP-1, mitogen-activated protein kinase phosphatase-1; MMP, matrix metalloproteinase; NDR1, N-myc downstream regulated 1; P4HA1, procollagen-proline, 2-oxoglutarate 4-dioxygenase (proline 4-hydroxylase), a1 polypeptide; PAS, Per-Arnt-Sim; PERK, PKR-like ER kinase; PPAR, peroxisome proliferators-activated receptor; PSMA3, proteasome (prosome,

macropain) subunit, a type 3; PTPN16, protein tyrosine phosphatase, non-receptor type 16; SFRS3, splicing factor, arginine ⁄ serine-rich 3 (SRp20); shRNA, short hairpin RNA; SUI1-RS1, suppressor of initiator codon mutations, related sequence 1; VEGF, vascular endothelial growth factor.

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Cellular oxygen is an important regulatory stimulus

for many physiological and pathological processes

Mammalian cells adapt to hypoxia by inducing the

expression of genes involved in anaerobic metabolism,

oxygen delivery and cell survival These diverse target

genes are induced by a common heterodimeric

transcription factor: hypoxia-inducible factor-a⁄ b

(HIF-a⁄ b) [1–4] The HIF-a and HIF-b subunits belong

to the basic helix–loop–helix (bHLH)-Per-Arnt-Sim

(PAS) protein family HIF-a is rapidly degraded in

normoxic cells, whereas HIF-b, also known as Arnt

(aryl hydrocarbon receptor nuclear translocator), is

con-stitutively expressed Under hypoxic conditions, HIF-a

is stabilized and translocates into the nucleus, where it

forms a heterodimer with the nuclear protein Arnt

Structural analyses of bHLH-PAS proteins have

deter-mined that interaction between the HLH-PAS domains

of each subunit mediates dimerization between HIF-a

and HIF-b, and that individual basic regions from

each protein interact with their corresponding DNA

elements Therefore, dimerization of bHLH-PAS

proteins is required for DNA binding [5]

The stability and activity of the a subunit are

inhib-ited by post-translational modification, specifically by

hydroxylation HIF-a hydroxylation is catalyzed by

HIF-a-specific proly-4-hydroxylase 2 and

HIF-a-spe-cific asparaginyl-hydroxylase, which utilize molecular

oxygen and a-ketoglutarate as cosubstrates The

hydroxylated proline residues (human HIF-1a Pro402

and Pro564) are recognized by the E3 ubiquitin ligase,

a von Hippel–Lindau protein which mediates HIF-1a

polyubiquitination and degradation by the 26S

protea-some [6,7] The hydroxylation of the human HIF-1a

asparagine residue 803 prevents HIF-a from recruiting

the CBP⁄ p300 coactivator A lack of oxygen has been

shown to reduce the activities of these two

oxygen-dependent hydroxylases, resulting in the stabilization

of the transactive form of HIF-1a [8,9]

HIF-1a was the first HIF-a isoform identified by

affinity purification, and HIF-2a (endothelial PAS

domain-containing protein 1) was later identified

through an homology search [10] Both HIF-1a and

HIF-2a form functional heterodimers with Arnt

Although knockout mice experiments have shown that

HIF-1a and HIF-2a have unique functions and are

non-redundant [11], no HIF-2a-specific target genes have

been identified HIF-1a and HIF-2a share a number of

target genes, but HIF-1a appears to be the predominant

form responsible for the induction of target genes [12]

Arnt was originally identified as a partner protein of

aryl hydrocarbon receptor (AhR) Similar to Arnt,

AhR also contains a bHLH-PAS domain at its

N-ter-minal domain Dioxin, an environmental pollutant, is

the most potent ligand for AhR Once bound to ligand, cytosolic AhR translocates into the nucleus and forms a heterodimer with Arnt Therefore, Arnt is a binding partner for both HIF-a and AhR [13,14] Pre-vious studies by Miller and Whitlock [15] led to the isolation of variant mouse hepa1c1c7 cell lines that lose responsiveness to dioxin using benzo(a)pyrene selection and fluorescence-activated cell sorting One of the variant cell lines, BpRc1, has normal AhR, but is defective in the nuclear localization of AhR Arnt transfection can complement this defect in BpRc1 cells, indicating that these variant cells are defective in Arnt [16,17] As Arnt is also required for the hypoxic induc-tion of HIF target genes, BpRc1 cells are also unre-sponsive to hypoxia, even in the presence of HIF-a Several studies have shown the role of Arnt in the basal expression of genes [18–20] Here, we emphasize the role of Arnt, especially in the hypoxic responses of gene expression By performing cDNA microarray analyses of hypoxic hepa1c1c7 cells and BpRc1 cells,

we identified both hypoxia-regulated genes and their Arnt dependence In addition, using HIF-1a knock-down cells, we investigated whether HIF-1a is required for the hypoxic responses of the identified genes

Results

Microarray analyses of hypoxia-regulated gene expression

We analyzed the changes in the expression of 5592 genes in response to hypoxic exposure (0.1% O2, 16 h) using Mouse 6K cDNA chips (TwinChip Mouse-6K) from Digital Genomics Inc (Seoul, South Korea) Mouse hepa1c1c7 and BpRc1 cells were exposed to hypoxia or normoxia (20% O2) for 16 h prior to RNA isolation and subsequent cDNA microarray analysis Four replicates were performed for each cell type using twin chips that incorporated dye-reversed hybridiza-tion Comparison of hepa1c1c7 and BpRc1 cells enabled us to investigate hypoxia-regulated gene expression, as well as the role played by HIF in the regulation of the hypoxic response Based on our anal-yses of 5592 genes, we selected statistically relevant genes with q values less than 0.1 for further analysis;

420 and 565 genes were selected from the analyses of hepa1c1c7 and BpRc1 cells, respectively Of these genes, 259 demonstrated q values of less than 0.1 in both analyses From the 259 genes, we selected hypoxia-induced genes that demonstrated a greater than 1.5-fold induction; 50 and 40 genes were selected from the analyses of hepa1c1c7 and BpRc1 cells, respectively (Tables 1 and S1) In addition, we selected

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Table 1 Hypoxia-induced genes identified by microarray analyses.

GenBank

accession

number

Gene

symbol Gene name

WT cells BpRc1 cells

Fold a q value b Fold a q value b Genes induced by hypoxia in both wild-type and BpRc1 cells

AI956848 BNIP3 c BCL2 ⁄ adenovirus E1B 19 kDa-interacting protein 3, NIP3 8.523 0.005 8.956 0.003 AI325917 PTPN16 c Protein tyrosine phosphatase, non-receptor type 16 6.409 0.005 2.264 0.003

AI323719 SUI1-RS1 c Suppressor of initiator codon mutations, related sequence 1

(Saccharomyces cerevisiae)

3.628 0.005 1.695 0.003

AI413228 – Mus musculus, clone MGC:18904 IMAGE:4240711, mRNA,

complete cds

2.168 0.005 1.637 0.003

AI504706 – ESTs, weakly similar to hair mouse hairless protein

(M musculus)

2.022 0.005 1.845 0.003

AI415729 – ESTs, moderately similar to sylm_human probable leucyl-tRNA

synthetase, mitochondrial precursor (Homo sapiens)

1.846 0.007 1.608 0.003 AW321053 AIRAP Arsenite inducible RNA-associated protein (Airap) 1.682 0.013 2.602 0.003

Genes induced by hypoxia in wild-type cells

AI323613 INPP5D Inositol polyphosphate-5-phosphatase, 145 kDa 3.271 0.005 1.357 0.072

AI323453 P4HA1 c Procollagen-proline, 2-oxoglutarate 4-dioxygenase

(proline 4-hydroxylase), a1 polypeptide

2.143 0.005 1.373 0.005

AI848411 BTG1 B-cell translocation gene 1, anti-proliferative 2.056 0.005 1.216 0.072 AI451895 RPGRIP1 Retinitis pigmentosa GTPase regulator interacting protein 1 1.838 0.005 1.269 0.051 AI452157 – ESTs, weakly similar to the KIAA0146 gene product is novel

(H sapiens)

1.837 0.005 1.427 0.005

AI842276 B3GALT2 UDP-Gal:betaGlcNAc beta 1,3-galactosyltransferase, polypeptide 2 1.666 0.005 1.323 0.016

AI452202 – DNA segment, Chr 12, Wayne State University 95, expressed 1.577 0.005 1.273 0.012

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genes that were repressed in response to hypoxia and

that demonstrated a less than 0.6-fold induction; 36

and 40 genes were selected from the analysis of

hepa1c1c7 and BpRc1 cells, respectively (Tables S1

and 3) Compared with northern analyses or

quantita-tive real-time reverse transcription-polymerase chain

reaction (Q-PCR), we found that the cDNA

micro-array analyses underestimated the fold change of gene

expression In order to take more genes into

consider-ation, we arbitrarily chose the values of 1.5- and

0.6-fold induction

Genes that are induced by hypoxia

The 50 genes that demonstrated a greater than

1.5-fold induction in hepa1c1c7 cells were considered to

be induced by hypoxia (Table 1) Of these 50 genes,

26 demonstrated a less than 1.5-fold induction in

BpRc1 cells, suggesting that these genes were induced

by hypoxia in an Arnt-dependent manner The

remaining 24 genes demonstrated a greater than

1.5-fold induction in both cell lines, suggesting that their

expression was regulated independent of Arnt To confirm these findings, nine of the 50 up-regulated genes were selected for Q-PCR analysis The fold induction of each gene in both hepa1c1c7 and BpRc1 cells was analyzed by one-way analysis of variance (ANOVA) (Table 2) Q-PCR analysis confirmed that each of the nine genes [suppressor of initiator codon mutations, related sequence 1 (SUI1-RS1), protein tyrosine phosphatase, non-receptor type 16 (PTPN16), N-myc downstream regulated 1 (NDR1), cyclin G2 (CCNG2), vascular endothelial growth factor (VEGF), BCL-2⁄ adenovirus E1B 19 kDa-interacting protein 3 (BNIP3), basigin (BSG), immediate early response 3 (IER3) and procollagen-proline, 2-oxoglut-arate 4-dioxygenase (proline 4-hydroxylase), a1 poly-peptide (P4HA1)] was induced by hypoxia with the indicated fold and P value (P < 0.05) in hepa1c1c7 cells (Fig 1 and Table 2) In contrast, Q-PCR analy-ses demonstrated that, in BpRc1 cells, the induction folds of eight genes (PTPN16, NDR1, CCNG2, VEGF, BNIP3, BSG, IER3 and P4HA1) were signifi-cantly lower, and their P values were greater than

Table 2 Hypoxia-induced gene expression analyzed by both microarray and Q-PCR up, hypoxia-induced expression pattern, fold ‡ 1.5; down, hypoxia-repressed expression pattern, fold < 0.6; nc, no change, 0.6 £ fold < 1.5; ns, not statistically significant, P value > 0.05; ), Arnt independent; +, Arnt dependent.

Gene

symbol

Arnt dependence

Arnt dependence

a Fold: a ratio of expression in hypoxia⁄ expression in normoxia b q value: significant difference, false discovery rate (FDR), selected q value < 0.1.

Table 1 Continued.

GenBank

accession

number

Gene

Fold a q value b Fold a q value b

AI324697 SNRPA Small nuclear ribonucleoprotein polypeptide A 1.519 0.025 1.449 0.005

a Fold: a ratio of expression in hypoxia⁄ expression in normoxia b q value: significant difference, false discovery rate (FDR), selected q value < 0.1 c Bold characters indicate genes that were further investigated by Q-PCR as reported in Fig 1.

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0.05, suggesting that hypoxia induced these eight

genes in an Arnt-dependent manner (Table 2)

Fur-thermore, Q-PCR analysis confirmed that SUI1-RS1

expression was significantly induced in both

hepa1c1c7 and BpRc1 cells, suggesting that expression

of SUI1-RS1 was regulated in an Arnt-independent

manner in response to hypoxia

Based on these Q-PCR results, we were able to con-firm the validity of our microarray analysis of hypoxia-induced gene expression Microarray analysis demonstrated that six genes (SUI1-RS1, PTPN16, NDR1, CCNG2, VEGF and BNIP3) were induced by hypoxia in both wild-type and Arnt-defective cells, and that an additional three genes (BSG, IER3 and

A

B

Hypoxia – + – +

Cell WT BpRc1

5

4

3

2

1

0

BSG mRNA

Hypoxia – + – + Cell WT BpRc1

120

100

80

60

40

20

0

PTPN16 mRNA

Hypoxia – + – + Cell WT BpRc1

25

20

15

10

5

0

VEGF mRNA

+ Hypoxia – + – Hypoxia – + – Cell WT BpRc1

25

20

15

10

5

0

P4HA1 mRNA

Cell

Hypoxia – + – +

Cell WT BpRc1

6

5

4

3

2

1

0

SUI1 -RS1 mRNA

Hypoxia – + – + Cell WT BpRc1

80

60

40

20

0

NDR1 mRNA

20

15

10

5

0

CCNG2 mRNA

Hypoxia – + – +

Cell WT BpRc1

Hypoxia – + – + Cell WT BpRc1

20

15

10

5

0

BNIP3 mRNA

Hypoxia Cell

– + – +

WT BpRc1

18

15

12

9

6

3

0

IER3 mRNA

Fig 1 mRNA levels of hypoxia-induced genes analyzed by Q-PCR (A, B) Wild-type (WT) hepa1c1c7 cells and BpRc1 cells were incubated

in hypoxic conditions for 16 h Total RNA was isolated and quantified by Q-PCR 18S rRNA expression levels were used for normalization Values are presented as the average ± standard deviation of three independent experiments Statistical analysis of the Q-PCR data was evaluated using one-way ANOVA.

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P4HA1) were not induced in Arnt-deficient cells

How-ever, Q-PCR analysis indicated that, with the

excep-tion of SUI1-RS1, eight of the nine genes were found

to be induced by hypoxia in an Arnt-dependent

man-ner Therefore, only four of these genes (SUI1-RS1,

BSG, IER3 and P4HA1) showed a consistent

expres-sion pattern in both Q-PCR and microarray analyses,

indicating that our microarray analysis was able to

predict Arnt-dependent expression of each gene in

response to hypoxia with a 44.5% (4⁄ 9) probability

Genes that are repressed by hypoxia The 36 genes that demonstrated a fold induction of less than 0.6 were considered to be repressed by hypoxia (Table 3) Of these, nine demonstrated a fold induction between 0.6 and 1.5 in BpRc1 cells, suggest-ing that they were repressed in an Arnt-dependent manner An additional 27 genes that demonstrated a fold induction of less than 0.6 in both cell lines were believed to be repressed in an Arnt-independent

Table 3 Hypoxia-repressed genes identified by microarray analyses.

GenBank

accession

number

Gene

symbol Gene name

Fold a q value b Fold a q value b Genes repressed by hypoxia in wild-type cells

AI324252 MAD2L1 c MAD2 (mitotic arrest deficient, homolog)-like 1 (yeast) 0.593 0.010 0.710 0.003

AI843948 PSMA3c Proteasome (prosome, macropain) subunit, a type 3 0.537 0.014 0.744 0.012 AI528616 SFRS3 c Splicing factor, arginine ⁄ serine-rich 3 (SRp20) 0.532 0.058 0.670 0.003

Genes repressed by hypoxia in both wild-type and BpRc1 cells

AI504950 ABCC3 c ATP-binding cassette, sub-family C (CFTR ⁄ MRP), member 3 0.599 0.029 0.426 0.003

AI853888 HIRIP5 Histone cell cycle regulation defective interacting protein 5 0.577 0.005 0.590 0.003

AI507479 – ESTs, moderately similar to Z277_human zinc finger

protein 277 (Homo sapiens)

AI853883 SHD src homology 2 domain-containing transforming protein D 0.484 0.005 0.471 0.003

AI504558 – Mus musculus, similar to hypothetical protein FLJ20174,

clone IMAGE:3595651, mRNA, partial cds

a Fold: a ratio of expression in hypoxia⁄ expression in normoxia b

q value: significant difference, false discovery rate (FDR), selected q value < 0.1 c Bold characters indicate genes that were further investigated by Q-PCR as reported in Fig 2.

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manner in response to hypoxia To validate these

results, eight of the 36 genes [MAD2 (mitotic arrest

deficient, homolog)-like 1 (yeast) (MAD2L1), heat

shock protein, 60 kDa (HSP60), FK506 binding

pro-tein 4 (59 kDa) (FKBP4), proteasome (prosome,

macropain) subunit, a type 3 (PSMA3), guanine

nucle-otide binding protein, a 11 (GNA11), splicing factor,

arginine⁄ serine-rich 3 (SRp20) (SFRS3), dual

specific-ity phosphatase 12 (DUSP12) and ATP-binding

cas-sette, subfamily C (CFTR⁄ MRP), member 3 (ABCC3)] were selected for Q-PCR (Fig 2) Our analysis deter-mined that four of the eight genes (MAD2L1, HSP60, FKBP4 and PSMA3) were significantly repressed in response to hypoxia as measured by Q-PCR analysis (P < 0.05) in hepa1c1c7 cells (Fig 2 and Table 4) Based on these results, we were able to demonstrate that our microarray analysis was able to predict hypoxia-repressed gene expression with a 50%

proba-A

B

2.0

1.5

1.0

0.5

0

DUSP12 mRNA

4

3

2

1

0

ABCC3 mRNA

2.5

2.0

1.5

1.0

0.5

0

MAD2L1 mRNA

1.6

1.4

1.2

1.0

0.8

0.6

0.4

0.2

0

PSMA3 mRNA

Hypoxia Cell

1.4 1.2 1.0 0.8 0.6 0.4 0.2 0

GNA11 mRNA

1.2 1.0 0.8 0.6 0.4 0.2 0

HSP60 mRNA

1.2 1.0 0.8 0.6 0.4 0.2 0

FKBP4 mRNA

2.5

2.0

1.5

1.0

0.5

0

SFRS3 mRNA

Fig 2 mRNA levels of hypoxia-repressed genes analyzed by Q-PCR (A,B) Wild-type (WT) hepa1c1c7 cells and BpRc1 cells were incubated

in hypoxic conditions for 16 h The expression level of each gene was quantified by Q-PCR Values are presented as the average ± standard deviation of three independent experiments Statistical analysis of the Q-PCR data was evaluated using one-way ANOVA.

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bility (4⁄ 8) in hepa1c1c7 cells We next compared the

fold induction of these eight genes in Arnt-defective

BpRc1 cells Microarray analyses showed that two

genes (DUSP12 and ABCC3) were repressed by

hypoxia in both hepa1c1c7 and BpRc1 cells, whereas

the remaining six genes (MAD2L1, HSP60, FKBP4,

PSMA3, GNA11 and SFRS3) were only repressed in

wild-type cells However, Q-PCR analyses indicated

that only two (MAD2L1 and HSP60) of the

four (MAD2L1, HSP60, FKBP4, and PSMA3)

confirmed genes were believed to be repressed in an

Arnt-dependent manner These results suggested that

our microarray analysis was able to predict

Arnt-dependent repression of each gene with a 50%

probability

Genes that are regulated by hypoxia in

a HIF-1a⁄ b-dependent manner

Q-PCR analyses confirmed that nine genes were

induced and four were repressed in response to

hypoxia (Tables 2 and 4), and that 10 of the 13

con-firmed genes were regulated by hypoxia in an

Arnt-dependent manner The remaining three genes

(SUI1-RS1, FKBP4 and PSMA3) were found to be

regulated in an Arnt-independent manner To

substan-tiate the specific role of Arnt, we used BpRc1 cells

infected with retrovirus expression full-length Arnt

[17] BpRc1 cells reconstituted with full-length Arnt

restored the hypoxia-induced (IER3, BSG, BNIP3,

VEGF, CCNG2, NDR1, P4HA1, PTPN16) or

hypoxia-repressed (HSP60, MAD2L1) gene expression

(Table 5), confirming that these 10 genes are regulated

by hypoxia in an Arnt-dependent manner The fold

induction of the genes was often greater in BpRc1 cells reconstituted with Arnt, reflecting that overexpression

of Arnt endowed the cells with increased responsibili-ties to hypoxia In addition, the Arnt-dependent induc-tion of five genes (IER3, BSG, BNIP3, VEGF and NDR1) and Arnt-dependent repression of two genes (MAD2L1 and HSP60) were validated by northern blot analysis (Fig 3)

Finally, we investigated the role of HIF-1a in regu-lating the expression of these genes in response to hypoxia by analyzing murine preadipoctyes 3T3-L1,

in which HIF-1a was knocked down by infection of retrovirus encoding a short hairpin RNA (shRNA) against HIF-1a Western blot analyses confirmed a specific reduction of HIF-1a protein by the cognate shRNA in 3T3-L1 cells (Fig 4A) Q-PCR analysis determined that IER3, BSG, BNIP3, VEGF, CCNG2 and NDR1 were induced in response to hypoxia in both an Arnt- and HIF-1a-dependent manner, indicat-ing that they are the target genes for HIF-1a⁄ b (Table 5 and Fig 4B,C)

Interestingly, it was determined that hypoxia induced both P4HA1 and PTPN16 in an Arnt-depen-dent, but HIF-1a-independent manner shRNA knockdown of HIF-1a failed to completely abolish hypoxia-induced expression of both P4HA1 and PTPN16, but instead reduced the fold induction of these genes by approximately one-half, thereby sug-gesting that HIF-1a plays a role in regulating hypoxia-mediated induction of these genes, at least in part (Table 5) As Arnt is a common binding partner for both HIF-1a and HIF-2a, HIF-2a may also play a partial role in regulating the hypoxia-dependent induc-tion of these genes

Table 4 Hypoxia-repressed gene expression analyzed by both microarray and Q-PCR up, hypoxia-induced expression pattern, fold ‡ 1.5; down, hypoxia-repressed expression pattern, fold < 0.6; nc, no change, 0.6 £ fold < 1.5; ns, not statistically significant, P value > 0.05; ), Arnt independent; +, Arnt dependent.

Gene

symbol

Arnt dependence

Arnt dependence

a Fold: a ratio of expression in hypoxia⁄ expression in normoxia b q value: significant difference, false discovery rate (FDR), selected q value < 0.1 c na, not applied; Arnt dependence was not applied when gene expression pattern of Q-PCR was different from microarray in hepa1c1c7 cells.

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Gene symbol

Arnt dependence

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In contrast, our results demonstrated that SUI1-RS1

expression was induced in response to hypoxia, even in

the absence of both Arnt and HIF-1a, suggesting that

neither HIF-1a nor HIF-2a was required for

hypoxia-induced expression of this gene Therefore, our results

demonstrated that HIF was not required for the

induc-tion of SUI1-RS1 in response to hypoxia In addiinduc-tion,

our results demonstrated that PSMA3 was repressed

by hypoxia in the absence of both Arnt and HIF-1a,

and that HSP60 was repressed by hypoxia in an

Arnt-and HIF-1a-dependent manner Finally, we determined

that MAD2L1 and FKBP4 were repressed in

hepa1c1c7 cells, but not in 3T3-L1 cells, suggesting

that these genes were repressed in response to hypoxia

in a cell type-specific manner

Discussion

In this study, we identified 259 hypoxia-regulated genes

by microarray analysis and confirmed the expression

profiles of 17 of these genes by Q-PCR analysis Col-lectively, we determined that 13 of the 17 genes (76.5%) were regulated, as predicted, by microarray analysis By comparing the results of our microarray analysis between wild-type cells and BpRc1 cells, we predicted the Arnt dependence of the confirmed 13 genes (Tables 2 and 4) Q-PCR analyses determined that only six of the 13 genes (46.2%) showed a consis-tent pattern of expression when compared with our microarray analysis for Arnt-dependent regulation in response to hypoxia

BNIP3, VEGF, CCNG2 and NDR1 have been iden-tified as HIF-1 target genes [21,22] However, the results of microarray analyses indicated that they were also induced in BpRc1 cells in response to hypoxia Compared with microarray analysis, it was determined that Q-PCR analysis of wild-type hepa1c1c7 cells resulted in a greater fold induction of these genes, sug-gesting that our microarray analysis quantitatively underestimated the changes in gene expression in wild-type cells In contrast, both microarray and Q-PCR analyses resulted in comparable levels of fold induction

in BpRc1 cells (Table 2) [19] However, the P value of fold induction measured by Q-PCR was determined to

be too large to accept the difference between the norm-oxic and hypnorm-oxic mRNA level of the gene in BpRc1 cells (P > 0.05) Therefore, Q-PCR analysis demon-strates that Arnt is important for the induction, but less necessary for the repression, of hypoxia-mediated gene expression when compared with predictions gen-erated by microarray analysis We next investigated whether the expression of these 13 genes was also regu-lated by HIF-1a (Table 5) It was determined that seven of the 13 genes (IER3, BSG, BNIP3, VEGF, CCNG2, NDR1 and HSP60) were regulated by hypoxia in both an Arnt- and HIF-1a-dependent manner Two genes (P4HA1 and PTPN16) were found

to be induced in response to hypoxia in an Arnt-dependent manner, but only partially regulated in a HIF-1a-dependent manner An additional two genes (SUI1-RS1 and PSMA3) were determined to be regu-lated under hypoxic conditions in both an Arnt- and HIF-1a-independent manner For the final two genes (MAD2L1 and FKBP4), it was determined that they were not repressed in 3T3-L1 cells, indicating that these genes are regulated in a cell type-specific manner

In addition to identifying previously known HIF-1 target genes, including BNIP3, VEGF, CCNG2, NDR1 and P4HA1 [21,22], and other known hypoxia-induc-ible genes, including IER3, BSG and PTPN16 [21–23], this report is the first to identify a number of novel hypoxia-regulated genes, including SUI1-RS1, HSP60 and PSMA3 (Table 5)

Hypoxia – + – + – +

BNIP3

NDR1

BSG IER3

VEGF

28S/18S

NB

α-Arnt

WB

MAD2L1 HSP60

28S/18S

Hypoxia – + – + – +

NB

A

B

Fig 3 Northern analyses of hypoxia-regulated genes (A,B)

Wild-type (WT) mouse hepa1c1c7 cells, the variant BpRc1 cells and

BpRc1 cells reconstituted with Arnt were exposed to hypoxic

condi-tions (0.1% O2) for 6 h Western blot analysis was performed using

30 lg of the cell lysates and HIF-1b (ARNT) antibody (top panel:

WB) For northern blot (NB) analysis, wild-type mouse hepa1c1c7

cells, the BpRc1 variant cell line and BpRc1 cells reconstituted with

Arnt were exposed to hypoxic conditions for 16 h; 20 lg of total

RNA from the treated cells was transferred onto a nitrocellulose

membrane Each blot was hybridized with the indicated [a- 32

P]-labeled probes Information on the probes is presented in Table S3.

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