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Báo cáo khoa học: E2A participates in a fine control of pre-mature B-cell apoptosis mediated by B-cell receptor signaling via transcriptional regulation of survivin, IAP2 and caspase-8 genes pot

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Based on these results, in this study, as a first step in elucidating the participation of B cell-specific factors in pre-mature B-cell apoptosis mediated by BCR stim-ulation by analyzing

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apoptosis mediated by B-cell receptor signaling via

transcriptional regulation of survivin, IAP2 and

caspase-8 genes

Kenji Toyonaga1,2,*, Hidehiko Kikuchi1,3,*, Koki Yamashita1, Masami Nakayama1, Kazuo Chijiiwa2 and Tatsuo Nakayama1,3

1 Section of Biochemistry and Molecular Biology, Department of Medical Sciences, Miyazaki Medical College, University of Miyazaki, Japan

2 Section of Surgical Oncology and Regulation of Organ Function, Department of Medical Science, Miyazaki Medical College, University of Miyazaki, Japan

3 Department of Life Science, Frontier Science Research Center, University of Miyazaki, Japan

It is widely known that B lymphocytes are essential to

immune responses in health and disease, and maintain

homeostasis by balancing cell viability and cell death

[1] The molecular mechanisms of B-lymphocyte

devel-opment have mostly been studied in mammalian bone

marrow and/or peripheral lymphoid tissue (e.g spleen)

Their development requires not only controlled

lineage-and locus-specific immunoglobulin gene recombination,

establishing unique antigen specificity of the B lympho-cytes, but also developmental stage-specific gene expres-sion participating in lymphoid cell proliferation and synthesis of immune mediators [1–3] In addition, vari-ous factors and/or signals control varivari-ous aspects of the normal development of B lymphocytes and func-tion of the immune system Such developmental activi-ties require numerous transcription factors, i.e the

Keywords

apoptosis; B cell; caspase; E2A; survivin

Correspondence

T Nakayama, Department of Life Science,

Frontier Science Research Center,

University of Miyazaki, 5200 Kihara,

Kiyotake, Miyazaki 889-1692, Japan

Fax: +81 985 85 6503

Tel: +81 985 85 3127

E-mail: tnakayam@med.miyazaki-u.ac.jp

*These authors contributed equally to this

work

(Received 8 May 2008, revised

16 December 2008, accepted 24

December 2008)

doi:10.1111/j.1742-4658.2009.06881.x

Antigen binding to the B-cell receptor (BCR) of pre-mature B lymphocytes induces their apoptotic cell death, but binding to the BCR of mature

B lymphocytes triggers activation and proliferation Binding to pre-mature

B lymphocytes is thought not only to function as a mechanism to exclude B-cell clones that possess the ability to react with self-antigen, but also to act as a defense mechanism in auto-immune diseases Cross-linking of BCR of pre-mature B-cell lines, including the chicken DT40 cell line, with anti-immunoglobulin IgG induces apoptotic cell death Treatment with phorbol 12-myristate 13-acetate/ionomycin, which mimics BCR stimula-tion, is used to study intracellular signal transduction of B lymphocytes Here, by analyzing the E2A-deficient DT40 cell line, E2A)/), we show that E2A deficiency prevents certain levels of apoptotic cell death mediated by BCR signaling In addition, E2A deficiency-linked BCR signaling controls the mimicked pre-mature B-cell apoptosis by PMA/ionomycin through ele-vated survivin plus inhibitor of apoptosis 2 levels, and reduced caspase-3 and caspase-8 activities, resulting in increased amounts of ICAD (inhibitor

of caspase-activated DNase), compared with those in the presence of E2A, followed by reduction of DNA fragmentation These findings will contrib-ute to the resolution of molecular mechanisms of negative selection of

B cells and also auto-immune diseases

Abbreviations

AIF, apoptosis-inducing factor; AKT, acutely transforming retrovirus AKT8 in rodent T cell lymphoma; BCR, B-cell receptor; CAD, caspase-activated DNase; EBF, early B-cell factor; FACS, fluorescence-caspase-activated cell sorter; GAPDH, glyceraldehyde 3-phosphate dehydrogenase; GATA-3, GATA binding protein-3; HAT, histone acetyltransferase; HDAC, histone deacetylase; IAP, inhibitor of apoptosis; PARP, poly(ADP-ribose)polymerase; PCAF, p300/CBP-associated factor; PMA, phorbol 12-myristate 13-acetate.

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basic helix-loop-helix transcription factors E2A, early

B cell factor (EBF), GATA-binding protein-3

(GATA-3), Pax5, PU.1, Ikaros and Aiolos, etc [2–4] Their

importance in B-cell development has been established

by knockout experiments on mouse hematopoietic stem

cells, in which all these functions are associated with

cessation of early stages of B-cell differentiation [3,5]

However, the expression of these factors is sustained

throughout development of lymphocytes in normal

mice after the observed block in their knockout mice

[6] Thus, their physiological functions beyond

develop-mental arrest remain poorly understood

Among these factors, E2A has been directly

impli-cated in transcriptional regulation of several B

lineage-specific genes, and has been shown to be essential for

Ig H- and L-chain recombination [5] Moreover, E2A

is required to initiate the expression of some B

lineage-specific genes such as EBF, mb-1 and B29, but not to

maintain expression of these genes [5] In general, E2A

has also been shown to promote proliferation and

sur-vival of various cell types [5,7] Recently, it was

revealed that E2A might be a key regulator of

apopto-sis versus proliferation in lymphoid cells, in addition

to its B lineage-determining function [8–11]

E2A-defi-cient mice develop T cell-derived lymphoma, and

enforced expression of an inhibitor of differentiation

proteins that indirectly inhibit E2A function [12,13] In

addition, ectopic expression of E47 or E12 induced

apoptotic cell death in thymic lymphoma cells derived

from E2A-deficient mice [14] These results indicate

that E2A might act as a tumor suppressor

It is also accepted that B lymphocytes are

suscepti-ble to receptor- and mitochondria-initiated cell death

at various stages of peripheral differentiation and

dur-ing immune responses [1,15,16] Recent genetic

evidence has contributed to understanding of the

BCR-dependent survival mechanism of mature B cells

[17,18] It is also known that E2A plays important

roles in the apoptosis of B lymphocytes [9–11] In

addition, Id3 protein, an E protein antagonist, induces

growth arrest and apoptosis in B-lymphocyte

progeni-tor cells [19] However, the impact of B cell-specific

factors, including E2A, on the BCR-mediated

apop-tosis of pre-mature B cells remains unclear, when

apoptosis is triggered by antigen stimulation

In order to better understand the roles of histone

acetyltransferases (HATs), histone deacetylases

(HDACs) and B cell-specific transcription factors in

B-cell functions, we have systematically generated

vari-ous homozygous mutants, including HDAC2)/),

GCN5)/), Aiolos)/) and E2A)/), using gene-targeting

techniques on the DT40 cell line, which was established

from chicken pre-mature B lymphocytes [20] Our

results show that HDAC2 controls the amount of IgM H-chain at two stages: transcription of its gene and alternative processing of its pre-mRNA [21] Recently,

we revealed not only that HDAC2 upregulates gene expression of EBF1, Pax5, Aiolos, Ikaros and HDAC7, and down-regulates those of E2A, p300/CBP-associated factor (PCAF), HDAC4 and HDAC5, but also that E2A upregulates expression of IgM H- and L-chain genes, but downregulates Aiolos, but Aiolos, EBF1, Pax5, and Ikaros downregulate expression of these two genes [22] These results, together with others, indicate that HDAC2 indirectly controls the expression of IgM H- and L-chain genes, through upregulated transcrip-tional regulation of EBF1, Pax5, Aiolos and Ikaros, and downregulated transcriptional regulation of E2A Moreover, our results indicated that GCN5 functions as

a supervisor in normal cell-cycle progression, with com-prehensive control over the expression of several cell cycle-related genes, as well as apoptosis-related genes, probably through alterations in the chromatin structure, indicated by the changing acetylation status of core histones surrounding these widely distributed genes [23] Recently, we showed that GCN5 and BCR signaling collaborate to induce apoptotic cell death of the DT40 cell line, through depletion of ICAD [inhibitor of caspase-activated DNase (CAD)] and inhibitor of apop-tosis 2 (IAP2), and activation of caspase activities [24] Based on these results, in this study, as a first step

in elucidating the participation of B cell-specific factors

in pre-mature B-cell apoptosis mediated by BCR stim-ulation by analyzing the E2A-deficient DT40 mutant E2A)/), we clarified the impact of E2A on apoptotic cell death of the DT40 cell line, and show that E2A is involved in fine control of pre-mature B-cell apoptosis mediated by BCR signaling, via transcriptional regula-tion of survivin, IAP2 and caspase-8 genes

Results

Insignificant influence of E2A deficiency on gene expression of apoptosis-related factors

To assess the influence of E2A deficiency on gene expression of apoptosis- and BCR signaling-related fac-tors, and other facfac-tors, we performed semi-quantitative RT-PCR on total RNAs prepared from DT40 and three independent E2A)/)clones (Fig 1) E2A deficiency did not have a significant influence on transcription of most

of these genes, except for survivin (to approximately 200%), PKCa (to approximately 60%), PKCg (to approximately 40%), PKCl (to approximately 40%) and PKCf (to approximately 160%) In addition, we performed immunoblot analyses to assess the influence

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of E2A deficiency on the amounts of proteins whose

mRNA levels were altered as noted above Consistent

with the results on mRNA levels, the protein levels for

survivin and PKCf were increased in E2A)/) and that

of PKCg was decreased (Fig S1); PKCa and PKCl

could not be detected using the available antibodies On

the other hand, transcription of various genes encoding

membrane-proximal factors, NF-jBs, transcription

fac-tors and B cell-related facfac-tors, amongst others, was not

altered in the E2A-deficient mutants (data not shown)

These insignificant effects of E2A deficiency on the

expression of numerous genes probably resulted in no

changes in the amount of apoptotic cells, as discussed

below (see Fig 2)

Moderate resistance to apoptosis is induced by

PMA/ionomycin in E2A

-/-We examined the influences of phorbol 12-myristate

13-acetate (PMA)/ionomycin and etoposide on

cell-cycle progression and proliferation of DT40 and E2A)/) cells Cells cultured in the presence of PMA/ ionomycin for 24 h or etoposide for 6 h were analyzed

by fluorescence-activated cell sorter (FACS) after stain-ing with propidium iodide (Fig 2A) As expected, eto-poside treatment caused apoptotic cell death for the two cell lines at the same level because the drug inhib-its the topoisomerase-2 activity that is essential for DNA replication Although PMA/ionomycin treatment

of DT40 cells resulted in cell-cycle distributions that were quite different from those in the case of the eto-poside treatment, it did induce apoptosis, and these findings agree with those reported previously [24] On the other hand, although PMA/ionomycin treatment

of E2A)/) did not alter the cell-cycle distribution pat-tern up to 24 h (cell growth was slightly delayed there-after), the depletion of E2A slightly prevented apoptotic cell death even in the presence of PMA/ ionomycin To confirm these findings, we examined the effects of PMA/ionomycin treatment on the viability

Fig 1 Effect of E2A deficiency on gene expression of apoptosis-related factors, caspases, caspase-regulating factors, CAD/ICAD and PKCs Total RNAs were extracted from DT40 and three independent E2A)/)clones (1–3), and mRNA levels were determined by semi-quantitative RT-PCR using appropriate primers The chicken GAPDH gene was used as an internal control The numbers under the panels indicate the number of cycles used for PCR.

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of DT40 and E2A)/)(Fig 2B) As expected, the

viabil-ity of the two cell lines did not differ in the absence of

PMA/ionomycin However, in the presence of PMA

and ionomycin, the viability of E2A)/) (approximately

50% at 48 h) was slightly higher than that of DT40,

which was dramatically reduced (approximately 20%

by 48 h) We undertook a comparative analysis of

changes in the morphological structure of nucleus as

an effect of PMA/ionomycin treatment in DT40 and

E2A)/) (Fig 2C) Nuclear fragmentation, another

characteristic of apoptosis, was partially hindered in

E2A)/), but it was clearly detected for DT40 in the

presence of PMA/ionomycin These results show that

E2A deficiency leads to blockage of the induced apop-totic cell death that is seen in the DT40 cell line when treated with PMA/ionomycin

Influence of depletion of B cell-specific transcrip-tion factors, HDACs and HATs on resistance to apoptosis induced by PMA/ionomycin treatment Using gene-targeting techniques, we systematically gen-erated several homozygous DT40 mutants that lacked genes encoding B cell-specific transcription factors, HDACs or HATs [20,22] Our previous results revealed that GCN5 and PMA/ionomycin treatment

C

Fig 2 Analyses of apoptosis in E2A)/) (A) Effects of PMA/ionomycin and etoposide treatments on cell-cycle distributions of DT40 and E2A)/) DT40 and E2A)/)cells treated with etoposide (10 lgÆmL)1) for 6 h, or with PMA (10 ngÆmL)1) plus ionomycin (1 l M ) for 24 h, were processed for DNA content analysis by propidium iodide staining Nuclei were analyzed by flow cytometry (FACSCalibur, Becton Dickinson and Company, Franklin Lakes, NJ, USA), and data for DT40 and E2A)/)(clone 1) were plotted on linear histograms as relative cell number (y axis) against red fluorescence intensity (x axis) The percentages of the various cell-cycle phases (sub-G1, G1, S and G2/M) for for DT40 and E2A)/)(clone 1), together with those of two other E2A)/)clones (2 and 3) are indicated in the table (B) Sensitivity of DT40 (circles) and E2A)/)(squares, triangles and diamonds) to PMA/ionomycin-mediated apoptotic cell death Cells were resuspended in DMEM containing 10% v/v fetal bovine serum, and treated with (filled symbols) or without (open symbols) 10 ngÆmL)1PMA plus 1 l M ionomycin at 37 C for

up to 48 h Viable cells were counted by the trypan blue dye exclusion method Data represent the mean of two separate experiments, and error bars indicate the standard deviation (C) Morphology of DT40 and E2A)/) (clone 1) cells treated with PMA/ionomycin Cells were cultured for 24 h without (no treatment) or with PMA/ionomycin (PMA/ionomycin), and their nucleus forms were analyzed by microscopy.

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cooperatively induce apoptotic cell death in the DT40

cell line [24] As a first step in elucidating the

participa-tion of E2A in apoptosis of DT40 cells, we examined

the effects of PMA/ionomycin treatment for these

homozygous DT40 mutants by FACS after staining

with propidium iodide and/or determination of cell

viability Detailed information on the generation of

mutants lacking Helios, Pax5, MORF, MOZ and

MOZ/MORF will be shown elsewhere As shown in

Table 1, depletion of HDAC2, as well as of E2A and

GCN5, prevented the apoptosis induced by

PMA/ion-omycin treatment, but depletion of Aiolos promoted

such apoptosis No changes were detected in the

remaining single mutants lacking EBF, Helios, Pax5,

HDAC1, HDAC7, SIRT1, SIRT2, PCAF, HAT1,

MORF or MOZ, or in a double knockout mutant

lacking MOZ and MORF These findings suggest that,

among the B cell-specific transcription factors, HDACs

and HATs examined, E2A, Aiolos, HDAC2 and

GCN5 preferentially participate in control of the

apop-totic cell death induced by PMA/ionomycin treatment

of the DT40 cell line

Up- and downregulation of expression of the E2A

or Aiolos genes by PMA/ionomycin treatment

To determine whether or not expression of the

B cell-specific transcription factors, HDACs and HATs

mentioned above is influenced by PMA/ionomycin treatment, DT40 and E2A)/)were cultured in the pres-ence of PMA/ionomycin, and RT-PCR was performed (Fig 3 and Fig S2) E2A deficiency dramatically decreased the mRNA level of Aiolos (to approximately 30%), but did not have any effect on transcripts of the remaining B cell-specific transcription factors, or HDACs and HATs On the other hand, PMA/ionomy-cin treatment dramatically altered gene expression of E2A in DT40 (to approximately 320% by 24 h) and that of Aiolos in both DT40 and E2A)/) (to less than 10% by 24 h) However, in the two cell lines, the treat-ment did not have significant effects on mRNA levels

of HDAC1, HDAC2, SIRT2, HAT1 and MORF, and slightly distinct but almost similar effects on mRNA levels of EBF, Pax5, HDAC7, SIRT1, GCN5, PCAF and MOZ These findings, together with those shown

in Table 1, indicate not only that HDAC2 and GCN5 are necessary for control of the apoptosis of the DT40 cell line mediated by PMA/ionomycin treatment, but also that their own transcription is not influenced by PMA/ionomycin Therefore, the apoptotic cell death

of DT40 mediated by PMA/ionomycin treatment must

be under the control of the elevated or decreased amounts of E2A or Aiolos

Upregulation of survivin and IAP2 gene expres-sion and no effect on caspase-8 gene expresexpres-sion

by PMA/ionomycin treatment in E2A

-/-To further clarify the molecular mechanism linked to the apoptotic induction of the DT40 cell line coopera-tively mediated by E2A and BCR signaling, DT40 and E2A)/)were cultured in the presence of PMA/ionomy-cin, and RT-PCR was performed for various factors (Fig 3 and Fig S2) E2A deficiency did not alter the gene expression of the apoptosis-related factors bcl-2, bcl-xL, bak, Apaf-1, cytochrome c, acutely transform-ing retrovirus AKT8 in rodent T cell lymphoma (AKT), apoptosis-inducing factor (AIF) and poly (ADP-ribose)polymerase (PARP) (also shown in Fig 1) The PMA/ionomycin treatment did not have a significant influence on the mRNA levels of bcl-2, bak, Apaf-1, cytochrome c, AKT and AIF, and had slightly distinct but almost similar effects on the mRNA levels

of bcl-xL and PARP in both DT40 and E2A)/) Similarly, depletion of E2A showed no effects on gene expression of CAD and ICAD (also shown in Fig 1) PMA/ionomycin treatment showed similar effects on the expression of CAD and ICAD genes in both DT40 and E2A)/), i.e the CAD mRNA level decreased by 3 h but thereafter increased to the con-trol level by 24 h, and the ICAD mRNA level

Table 1 Influences of depletion of B cell-specific transcription

fac-tors, HDACs and HATs on resistance to apoptosis induced by

PMA/ionomycin treatment.

Resistance for PMA/ionomycin

B cell-specific factors

HDACs

HATs

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decreased by 3 h and thereafter remained unchanged

at 24 h in the presence of PMA/ionomycin These

findings indicate that E2A and BCR stimulation have

no effects on gene expression of CAD and ICAD, in

contrast to the effects of GCN5 and BCR

stimula-tion [24]

With regard to caspases, E2A depletion showed no

effects on expression of 3, 6,

caspase-8, caspase-9 and caspase-10 genes (also shown in

Fig 1) On the other hand, in both DT40 and E2A)/),

the caspase-6 mRNA level was decreased gradually

by PMA/ionomycin treatment by 24 h, and the

cas-pase-10 mRNA level was increased by 3 h and

there-after decreased dramatically by 24 h Expression of

caspase-3 and caspase-9 remained unchanged in the

presence of PMA/ionomycin Interestingly, in DT40,

PMA/ionomycin treatment increased the caspase-8

mRNA level by 3 h (to approximately 160%) and

this level remained unchanged at 24 h, but the treat-ment showed no change in the transcript level of caspase-8 in E2A)/) These findings indicate that expression of most caspase genes is not much influ-enced by either E2A or BCR stimulation, except that

of caspase-8

With regard to caspase-regulating factors, depletion

of E2A increased transcription of the survivin gene (to approximately 220%), but did not have a significant effect on expression of the FLIP, IAP1, IAP2 and Smac genes However, PMA/ionomycin treatment had distinct effects on expression of these caspase inhibi-tors The IAP1 or Smac mRNA levels increased or decreased slightly by 3 h and thereafter remained unchanged in both DT40 and E2A)/) The FLIP mRNA level was slightly decreased at 3 h (to approxi-mately 60%) and thereafter increased to the control level in DT40, but remained unchanged in E2A)/)

Fig 3 Effects of PMA/ionomycin treatment on gene expression of B cell-specific factors, HDACs, HATs, apoptosis-related factors, CAD/ ICAD, caspases and caspase-regulating factors Total RNAs were extracted from PMA/ionomycin-treated DT40 and E2A)/)(clone 1) at indi-cated times up to 24 h, and then the mRNA levels of appropriate genes were determined by RT-PCR The chicken GAPDH gene was used

as a control.

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Interestingly, whereas the IAP2 mRNA level in DT40

was significantly decreased by PMA/ionomycin

treat-ment at 3 h (to approximately 30%) and thereafter

remained unchanged, the transcript level in E2A)/)

was unchanged at 3 h, and thereafter decreased by

24 h (to approximately 20%) Furthermore, in E2A)/),

the survivin mRNA level was maintained at a high

level (approximately 220%) when treated with PMA

and ionomycin for 3 h, and thereafter decreased to

slightly higher level than that in DT40; the mRNA

level was not influenced by the drug treatment in

DT40 These findings indicate not only that E2A

downregulates transcription of the survivin gene and

has no effects on that of FLIP, IAP1, IAP2 or Smac,

but also that BCR stimulation and E2A cooperatively

control expression of FLIP, IAP2 and survivin genes

Resistance to PMA/ionomycin-mediated

apoptosis of E2A-/-is brought about by

increased amounts of survivin and IAP2, and

reduced activity of caspase-3

Next we examined the effect of PMA/ionomycin

treat-ment on cellular protein levels of survivin, IAP2 and

ICAD, which are proximal factors controlling CAD

activity for DNA fragmentation, by immunoblotting

using their specific antibodies (Fig 4A) E2A deficiency

increased the protein levels of survivin (to

approxi-mately 190%), but had no effect on those of IAP2 and

ICAD Consistent with previous results [24],

PMA/ion-omycin treatment in DT40 dramatically decreased the

protein levels of IAP2 and ICAD (to approximately

40% and less than 10%) by 24 h, but had a moderate

influence on that of survivin (approximately 60% at

24 h) Therefore, in DT40 cells treated with

PMA/iono-mycin, the time courses of alterations in the protein and

mRNA levels of survivin were virtually similar, and the

gradual reductions in protein levels of IAP2 plus ICAD

compared with acute decreases (unchanged thereafter)

of their mRNA levels agreed with previous results [24]

On the other hand, in E2A)/), PMA/ionomycin

treatment did not influence the protein level of survivin

by 8 h and thereafter it decreased gradually

PMA/ion-omycin treatment did not change the protein level of

IAP2 Further, the PMA/ionomycin treatment showed

a moderate reduction in the protein level of ICAD (to

approximately 40%) by 24 h in E2A)/), i.e the rate of

decrease in the amount of ICAD in the mutant was

slower than that in DT40 Thus, in E2A)/), in the

presence of PMA/ionomycin, the time courses of

alter-ations in the mRNA and protein levels of survivin

were virtually similar up to 24 h, but the findings that

the protein levels of IAP2 or ICAD were increased or

slightly decreased by 24 h did not agree with the find-ings that the mRNA levels of IAP2 and ICAD were reduced slowly by 24 h or quickly by 3 h, respectively

To resolve this discrepancy between the results regarding the mRNA and protein levels of ICAD (and also IAP2), we next examined the effects of PMA/ ionomycin treatment on caspase activities (Fig 4B) Interestingly, in E2A)/), PMA/ionomycin treatment showed slightly decreased activities of caspase-3 (to approximately 50% by 16 h), caspase-8 (to approxi-mately 70% by 16 h) and caspase-9 (to approxiapproxi-mately 70% by 16 h), compared with those in DT40, probably due to a balance of the amounts of each of the three caspases and the inhibitors survivin and IAP2 (and also FLIP and IAP1) The slightly decreased caspase-8 activity mediated by PMA/ionomycin treatment in E2A)/) may have resulted from the balanced mRNA (and probably protein) levels of caspase-8, which lead

to activation of pro-caspase-9, causing formation of the active form of caspase-3, and of the inhibitors FLIP (for caspase-8), survivin (for caspase-9) and IAP2 (for caspase-3) [25] In E2A)/), the decreased activity of caspase-3 mediated by PMA/ionomycin treatment must depend on both the reduced amount of activated caspase-3 itself as a result of decreased caspase-8 (and probably caspase-9) activity, and the elevated protein (and also mRNA) levels of the inhibitors survivin and IAP2 As a result, the slow diminution of the protein level of ICAD by PMA/ionomycin treatment in E2A)/) (Fig 4A) must be due to its slight degradation medi-ated by suppressed caspase-3 activity, although the ICAD mRNA level was decreased when exposed for

3 h but thereafter remained unchanged, as did that in DT40 (Fig 3 and Fig S2)

Because the alterations in the mRNA level of CAD were the same in both DT40 and E2A)/) (Fig 3 and Fig S2), and its protein could not be detected by the available antibodies to assess CAD activity, we exam-ined the effects of PMA/ionomycin treatment on DNA fragmentation, a typical result of CAD activity (Fig 4C) The DNA fragmentation was found to be more moderate for E2A)/) than that observed for DT40, even in the presence of PMA/ionomycin up to

24 h These results indicate that the CAD activity in E2A)/) is suppressed by the moderately reduced amount of its inhibitor ICAD compared with that in DT40, resulted in decreased DNA fragmentation, a characteristic of apoptosis

Discussion

In recent years, numerous studies have been performed

to determine the physiological target genes of E2A

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B

C

Fig 4 Analyses of mechanisms of the resistance to PMA/ionomycin-mediated apoptosis in E2A)/) (A) Effects of PMA/ionomycin treatment

on protein levels of survivin, IAP2 and ICAD Whole proteins were isolated from PMA/ionomycin-treated DT40 and E2A)/)at indicated times

up to 24 h, and subjected to SDS–PAGE followed by immunoblotting Antibody binding was detected using secondary antibodies conjugated

to horseradish peroxidase, and then data analysis was performed using a luminescent image analyzer Left panel: typical immunoblot pattern (DT40 and E2A)/)clone 1) b-actin was used as a control The apparent molecular masses of marker proteins are indicated Right panel: time courses of protein levels for survivin, IAP2 and ICAD after treatment with PMA/ionomycin in DT40 (circles) and three E2A)/)clones (1–3) (squares, triangles and diamonds) Data are expressed as percentages of the control (DT40 at 0 h) (B) Effects of PMA/ionomycin treatment

on caspase activities Cell lysates were prepared from PMA/ionomycin-treated DT40 (circles) and E2A)/)clones (1–3) (squares, triangles and diamonds) at indicated times up to 16 h, and then caspase activity assays were performed using appropriate caspase assay kits Absorbance

at 405 nm was measured to determine activities Data represent the mean of two separate experiments, and error bars indicate standard deviation (C) Effects of PMA/ionomycin treatment on DNA fragmentation in DT40 and E2A)/) DNA was isolated from DT40 and E2A)/) cells incubated for 0, 8, 16 and 24 h in the presence of PMA and ionomycin, and analyzed by 1.5% agarose gel electrophoresis The sizes

of k-DNA digested with HindIII are indicated in kb Left panel: typical electrophoregram of DNA extracted from PMA/ionomycin-treated DT40 and E2A)/)(clone 1) Right panel: electrophoregram of DNA extracted from PMA/ionomycin-treated DT40 and three E2A)/)clones (1–3)

at 16 h.

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E2A directly activates the EBF gene [26] and regulates

expression of several genes, i.e k5, Rag-1, Vj1 and jo,

that are involved in D-JH rearrangement, cell survival,

Igj rearrangement, etc [26,27] E2A directly controls

IgH gene expression, and is involved in repressing the

Nfil3 and FGFR2 genes in pre-mature B lymphocytes

[28] Thus, E2A is one of the most essential regulators

at multiple stages of B-cell development In T

lympho-cytes, PLCc2, Cdk6, CD25, Tox, Gadd45a, Gadd45b,

Gfi1, Gfi1b, Socs1, Socs3, Id2, Eto2, Xbp1 etc have

been identified as novel E47 target genes using an

E2A-deficient lymphoma cell line [29] Recently, we reported

that apoptosis of the chicken DT40 cell line, a

pre-mature B-cell line, is cooperatively controlled by GCN5

and BCR stimulation via complex transcriptional

regulation of a number of genes encoding BCR

signal-ing-related factors, B cell-specific factors, transcription

factors and apoptosis-related factors, indicating that

both are necessary for apoptosis of DT40 cells [24] In

DT40 cells, BCR signaling is transduced from BCR

and membrane-proximal factors (Syk, BTK, BLNK

and PLCc2, etc), via mainly PKCd, PKCe and PKCf,

to NF-jBs (probably c-Rel and NFp50) This activated

signal mediated by BCR signaling is probably

trans-ducted separately into two apoptotic pathways, i.e

direct transduction of the signal into the CAD/ICAD

system and transduction of the activated signal into the

caspase cascade pathway However, understanding of

the participation of most B cell-specific factors in the

apoptotic process has remained elusive

Lack of E2A partially prevents the apoptotic cell

death seen in DT40 cells treated with PMA/ionomycin,

which mimics BCR stimulation (Fig 2); such apoptosis

is completely prevented by either GCN5 deficiency [24]

or HDAC2 deficiency, and is significantly accelerated

by Aiolos deficiency (Table 1) By analyzing E2A)/), we

revealed that E2A upregulates the expression of PKCa,

PKCg and PKCl genes, and downregulates the

expres-sion of survivin and PKCf genes, among the numerous

factors examined (Fig 1 and unpublished data) PMA/

ionomycin treatment increased expression of the E2A

gene and dramatically suppressed that of the Aiolos

gene in DT40, but in E2A)/)had no effects or similar

effects on the expression of other disrupted genes

(Fig 3A) and genes encoding B cell-specific factors,

HDACs and HATs (our unpublished data) These

results suggest not only that, among the B cell-specific

factors tested, E2A or Aiolos participates preferentially

in suppression or acceleration of apoptosis of the DT40

cell line, but also that, among the HAT and HDAC

families tested, gene expression of GCN5 and HDAC2,

which are essential for apoptotic cell death, are not

influenced directly by PMA/ionomycin treatment On

the other hand, in E2A)/), the detected protein level of Aiolos, which was already suppressed to a very low level, is further reduced by the treatment, resulting in suppression of apoptotic cell death compared with that

of DT40

The alterations in expression of E2A and Aiolos (and probably others) in DT40 when exposed to PMA/ionomycin accompany altered expression of vari-ous genes encoding apoptosis-related factors, caspases, caspase-regulating factors and CAD/ICAD (Fig 3 and Fig S2), and almost all of these results agreed with those in a previous report [24] In E2A)/), the altered expression of Aiolos (and probably others) in the presence of PMA/ionomycin results in (and/or accom-panies) slightly different effects on expression of the genes mentioned above, i.e the influences of PMA/ion-omycin treatment on expression of the survivin and IAP2 (and probably FLIP) genes were more moderate

in the mutant than in DT40, and no effect was observed on expression of caspase-8 However, in DT40, the PMA/ionomycin-induced alterations in gene expression of various B cell-specific factors, HDACs, HATs, apoptosis-related factors, caspases, caspase-reg-ulating factors and CAD/ICAD led to changes in the activities of caspase-3, caspase-8 and caspase-9, and in the protein levels of IAP2 and ICAD (Fig 4A,B), con-sistent with previous results [24] Interestingly, in E2A)/), the noticeable alterations in gene expression of survivin and IAP2 accompanied by unchanged gene expression of caspase-8 (and also various B cell-specific factors, HDACs, HATs, apoptosis-related factors, caspases, caspase-regulating factors and CAD/ICAD) mediated by PMA/ionomycin results in suppression of activities of caspase-3, caspase-8 and caspase-9, and alterations in the protein levels of survivin, IAP2 and ICAD (and probably FLIP) (Fig 4A,B)

Finally, in E2A)/), the slightly suppressed degrada-tion of ICAD molecules as an effect of reduced cas-pase-3 activity reduces CAD activity, leading to moderate fragmentation of DNA molecules (Fig 4C) Thus, progress towards apoptotic cell death in E2A)/)

is suppressed by collaboration of both BCR signaling and E2A depletion, mainly via moderate changes in amounts of the inhibitors survivin, IAP2 and ICAD (and probably FLIP) Thus, E2A is involved in fine control of pre-mature B-cell apoptosis mediated by BCR signaling via transcriptional regulation of survi-vin, IAP2, FLIP and caspase-8 genes

The observations in this study regarding the par-ticipation in apoptosis of Aiolos and HDAC2, which are now being further studied by us, as well as that

of GCN5, amongst others, will be useful in elucidat-ing not only the linkage between BCR signalelucidat-ing and

Trang 10

apoptosis cascades in pre-mature B-cell lines, but

also the molecular mechanism of negative selection

or development of B lymphocytes through cross-talk

among B-cell signaling, B cell-specific transcriptional

regulation and epigenetic chromatin topology

altera-tions These results, combined with other findings

obtained in the future, may contribute to clinical

understanding of auto-immune diseases and B-cell

lymphomas

Experimental procedures

Materials

PMA was purchased from Calbiochem (Darmstadt,

Ger-many) and ionomycin was purchased from Sigma (St Louis,

MO, USA) The antibodies used were anti-ICAD (Santa

Cruz Biotechnology Inc., Santa Cruz, CA, USA),

anti-cIAP-2 (Chemicon, Temecula, CA, USA), horseradish

per-oxidase-conjugated goat anti-rabbit immunoglobulin and

horseradish peroxidase-conjugated rabbit anti-mouse

immu-noglobulin (Dako Inc., Glostrup, Denmark)

Cell cultures and apoptosis induction

DT40 cells and all subclones were cultured essentially as

described previously [24] Apoptosis was induced as follows:

cells (2· 106

) in 10 mL of culture medium were incubated

with 10 ngÆmL)1 PMA plus 1 lm ionomycin or with

10 lgÆmL)1 etoposide at 37C Viable cells were counted

by the trypan blue dye exclusion method Flow cytometric

analyses, morphological analyses, the caspase activity assay

and the DNA fragmentation assay were performed as

described previously [24,30]

Semi-quantitative RT-PCR

Total RNAs were isolated from DT40 and its subclones

Reverse transcription was performed using a first-strand

DNA synthesis kit (Toyobo, Osaka, Japan) at 42C for

20 min, followed by heating at 99C for 5 min PCRs were

performed as described previously [24] using sense primers

and antisense primers synthesized according to the EST

data deposited in GenBank for the appropriate genes, and

listed in previous reports [22–24], except for SIRT1 (sense

primer 5¢-CTGTTTTTACCACCAAATCG-3¢ and antisense

primer 5¢-CAACTTGTTGCTTGTTGGAT-3¢) and SIRT2

(sense primer 5¢-ATGTCCCTCATGGGCTTCGG-3¢ and

antisense primer 5¢-TCACGGCTCTTTGTCGTCCC-3¢)

The chicken glyceraldehyde 3-phosphate dehydrogenase

(GAPDH) gene was used as an internal control PCR

prod-ucts were subjected to 1.5% agarose gel electrophoresis,

and analyzed using an LAS-1000plus luminescent image

analyzer (Fujifilm, Tokyo, Japan)

Immunoblotting Cells were treated with 10% trichroloacetic acid, collected

by centrifugation 20 000 g for 5 min at 4C, dissolved in 0.5 m Tris/HCl (pH 6.8) containing 2.5% SDS, 10% glyc-erol and 5% 2-mercaptoethanol, and heated at 100C for

5 min Immunoblotting was performed as described previ-ously [24] b-actin was used as a control

Acknowledgements

We thank Y Takami and H Suzuki for technical sup-port and H K Barman for editorial reading of the manuscript This work was supported in part by a Grant-in-Aid for Scientific Research from the Ministry

of Education, Culture, Sports, Science and Technology

of Japan

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