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Bcl-2 / E1B-19K-interacting protein 3 is a mediator ofcaspase-independent neuronal death in excitotoxicity Zhengfeng Zhang1,2, Ruoyang Shi1, Jiequn Weng1, Xingshun Xu3, Xin-Min Li1, Tian

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Bcl-2 / E1B-19K-interacting protein 3 is a mediator of

caspase-independent neuronal death in excitotoxicity

Zhengfeng Zhang1,2, Ruoyang Shi1, Jiequn Weng1, Xingshun Xu3, Xin-Min Li1, Tian-ming Gao4 and Jiming Kong1,4

1 Department of Human Anatomy and Cell Science, University of Manitoba, Winnipeg, Manitoba, Canada

2 Department of Orthopedics, Xinqiao Hospital, TheThrid Military Medical University, Chongqing, China

3 Institute of Neuroscience, Soochow University, Suzhou, Jiangsu Province, China

4 Department of Anatomy and Neurobiology, Southern Medical University, Guangzhou, China

Introduction

Excessive activation of glutamate receptors results in

excitatory neuronal cell death, a process called

excito-toxicity, which has been shown to be a contributory

factor to neuronal cell loss in neurodegenerative

diseases [1,2] The mechanisms responsible for

neuro-excitotoxicity include neuronal Ca2+overload [3,4],

mitochondrial depolarization [3,5–8], and opening of

mitochondrial permeability transition pores, through which mitochondrial solutes with molecular masses up

to 1.5 kDa can pass [9]

Members of the Bcl-2 family are important regulators

of apoptotic cell death [10–12] Antiapoptotic members

of the Bcl-2 family, including Bcl-2 and Bcl-XL, prevent apoptosis by preserving mitochondrial integrity [11]

Keywords

apoptosis; Bcl-2 ⁄ E1B-19K-interacting

protein 3 (BNIP3); caspase-independent

cell death; excitotoxicity; neuron

Correspondence

J Kong or T Gao, Department of Human

Anatomy and Cell Science, University of

Manitoba, 730 William Avenue, Winnipeg,

Manitoba R3E 0W3, Canada; Department of

Anatomy and Neurobiology, Southern

Medical University, Guangzhou 510515,

China

Fax: +1 204 789 3920

Tel: +1 204 977 5601; +011 86 20 6164 8216

E-mail: kongj@cc.umanitoba.ca;

tianminggao@tom.com

(Received 3 June 2010, revised 1

September 2010, accepted 25 October

2010)

doi:10.1111/j.1742-4658.2010.07939.x

Caspase-independent neuronal death has been shown to occur in neuroexci-totoxicity Here, we tested the hypothesis that the gene encoding

Bcl-2⁄ E1B-19K-interacting protein 3 (BNIP3) mediates caspase-independent neuronal death in excitotoxicity BNIP3 was not detectable in neurons under normal condition BNIP3 expression was increased dramatically in neurons in both in vivo and in vitro models of excitotoxicity Expression of full-length BNIP3 in primary hippocampal neurons induced atypical cell death that required protein synthesis but was largely independent of caspase activities Inhibition of BNIP3 expression by RNA interference protected against glutamate-induced neuronal cell death Thus, BNIP3 activation and expression appears to be both necessary and sufficient for neuronal apoptosis in excitotoxicity These results suggest that BNIP3 may

be a new target for neuronal rescue strategies

Abbreviations

BNIP3, Bcl-2 ⁄ E1B-19K-interacting protein 3; CL, contralateral; CNQX, 6-cyano-7-nitroquinaloxine-2,3-dione; EGFR, enhanced green

fluorescent protein; GST, glutathione-S-transferase; KA, kainic acid; NMDA, N-methyl- D -aspartate; RNAi, RNA interference; TUNEL, terminal deoxynucleotidyl transferase dUTP nick end labeling.

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Upon activation by death stimuli, proapoptotic

mem-bers of the Bcl-2 family, including Bad, Bax, Bid,

and Bim, permeabilize mitochondrial membranes [13]

Bcl-2⁄ E1B-19K-interacting protein 3 (BNIP3) is a

member of a unique subfamily of death-inducing

mito-chondrial proteins [14,15] BNIP3-induced cell death

has been characterized by early plasma membrane and

mitochondrial damage independently of cytochrome c

release and caspase activity [16,17] However, the extent

to which BNIP3 is involved in excitotoxicity-induced

neuronal cell death is not known Here, we tested the

hypothesis that BNIP3 is a gene that mediates

caspase-independent neuronal death in excitotoxicity Our

results show that BNIP3 expression is upregulated in

in vivo and in vitro models of neuroexcitotoxicity, that

expression of full-length BNIP3 induced an atypical

form of cell death, and that inhibition of BNIP3 by

RNA interference (RNAi) and expression of a

domi-nant negative form of BNIP3 that lacks the functional

transmembrane domain protected against

glutamate-induced neuronal cell death Thus, BNIP3 activation

and expression appear to be both necessary and

suffi-cient for atypical neuronal apoptosis in excitotoxicity

Results

Kainic acid (KA) is a specific agonist for the kainate

receptor (a subtype of the ionotrophic glutamate

recep-tor) that mimics the effect of glutamate Of the 15 rats

that received KA injections, five were used for

prepa-ration of brain sections and 10 for biochemical

analy-sis Under control conditions, wer and others were

able to only barely detect BNIP3 in brain tissue or

hippocampal neurons [17,18] As a first step to testing

the hypothesis that BNIP3 expression plays an

impor-tant role in neuroexcitotoxicity, we examined levels of

BNIP3 expression by immunohistochemistry in brains

of rats injected intrastriatally with KA Two days after

unilateral injection of KA, BNIP3-immunopositive

neurons were present in striatal areas adjacent to the site

of injection (Fig 1A) High levels of BNIP3

immuno-staining were found in the cytoplasm of striatal neurons

affected by the KA, and almost all of the

BNIP3-positive neurons showed signs of DNA damage when

stained with Hoescht 33342 (Fig 1B)

BNIP3-immuno-negative neurons showed normal nuclear morphology

DNA fragmentation in KA-induced neuronal cell

death was further confirmed by terminal

deoxynucleot-idyl transferase dUTP nick end labeling (TUNEL),

with TUNEL-positive nuclei being detected only in

areas adjacent to sites of KA injection, and not in the

contralateral (CL) striatum (Fig 1C,D) To confirm

that the increased expression of BNIP3 after KA

administration was caused by activation of kainate receptors, brain tissue was processed from rats that received intrastriatal injections (1 lL) of 2.5 nmol of

KA, 5 nmol of 6-cyano-7-nitroquinaloxine-2,3-dione (CNQX), a mixture of 5 nmol of CNQX and 2.5 nmol

of KA, or 50 mm Tris⁄ HCl (pH 7.4) BNIP3 expression was observed only in those rats that received KA alone, and not in those rats receiving CNQX or the buffer (data not shown)

To more quantitatively determine the levels of BNIP3 and determine the molecular mass of the BNIP3 expressed, immunoblots were run for samples derived from KA-injected striata, CL uninjected

striat-a, Tris⁄ HCl-injected striata and CL uninjected striata from Tris⁄ HCl-injected rats A 60 kDa band was present in KA-injected striata (Fig 1E); this band was much weaker in CL striata, and was absent in sam-ples from Tris⁄ HCl-injected rats To demonstrate the specificity of the BNIP3 immunoblotting, control experiments were performed in which the BNIP3 anti-body was first incubated for 30 min with a BNIP3– glutathione-S-transferase (GST) protein As shown in Fig 1E, immunoblotting for BNIP3 was completely blocked by the BNIP3–GST protein A nonspecific

62 kDa band was detected in all of the striatal sam-ples Quantification of the bands with the b-actin bands as internal controls revealed that injection of

KA upregulated BNIP3 expression nine-fold (Fig 1F;

n= 6)

To determine whether KA increased BNIP3 tran-scription as well as translation as described above, brain samples from KA-injected rats were processed

by in situ hybridization with an RNA probe specific for BNIP3 Levels of BNIP3 mRNA were increased by

KA (Fig 1G,H) Positive hybridization signals were found in a group of striatal neurons adjacent to the site of KA injection, whereas neurons in other brain areas showed very low levels of BNIP3 mRNA

To determine the mechanisms by which BNIP3 expression induced by excitotoxicity kills neurons, pri-mary cultures of rat hippocampal neurons were treated with glutamate for 6 h, maintained in Neurobasal medium for 24 h, and stained with trypan blue for membrane integrity As expected, glutamate increased neuronal cell death in a dose-dependent manner (Fig 2A); 70% of cells stained positively for trypan blue with 100 lm glutamate, and 10 lm glutamate killed 40% of hippocampal neurons Expression of BNIP3 was not detectable in the majority of untreated neurons, and less than 15% of the untreated neurons expressed low levels of BNIP3 according to immuno-histochemistry (Fig 2B) In contrast, more than 70%

of cells treated with 100 lm glutamate stained

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positively for BNIP3 (Fig 2C) Nuclei in BNIP3-positive

neurons showed a characteristic dysmorphic appearance

(Fig 2D) To determine the time course of BNIP3

expression, protein samples prepared from hippocampal

neurons were immunoblotted with an antibody against

BNIP3 A sample prepared from HEK 293 cells that

were transfected with T7-tagged pcDNA3–hBNIP3 was

included as a positive control As shown in Fig 2E,F,

levels of BNIP3 were significantly increased in neurons

after exposure to 100 lm glutamate for 36 h, and

peaked (seven-fold) at 60 h

Next, we determined the extent to which BNIP3

expression was necessary and sufficient to kill neurons

Primary cultures of hippocampal neurons at day 4 in culture were transfected using LipofectAMINE 2000 with a pcDNA3–hBNIP3 plasmid encoding full-length BNIP3, a pcDNA3–hBNIP3)163 plasmid encoding the first 163 amino acids of BNIP3, or the empty pcDNA3 plasmid The transfection efficiency was about 2–8%,

on the basis of immunohistochemistry with an anti-body against T7 that recognizes the T7 epitope tag Transient transfection with pcDNA3–hBNIP3 but not with pcDNA3–hBNIP3)163 (truncated BNIP3) resulted

in DNA condensation and neuronal cell death (Fig 3) The truncated BNIP3 was diffusely distributed in the cytoplasm, owing to the lack of its transmembrane

G

40

H

0 4 6 8 10

12

BNIP3

BNIP3 antibody

BNIP3 antibody +BNIP3–GST

62 kDa

BNIP3

(fold) **

**

KA CL Ctrl

KA CL Ctrl

β-actin

Fig 1 BNIP3 expression inbrain increased with excitotoxicity and correlated with mea-sures of ‘apoptotic’ cell death (A) BNIP3-immunopositive neurons were present adjacent to sites of KA injection The arrow points to the site of injection (B) DNA frag-mentation was observed in immuno-positive neurons (arrows)

BNIP3-immunonegative neurons showed normal nuclear morphology (arrowheads) (C) ‘Apop-totic’ nuclei, as detected by TUNEL labeling, surrounded sites of KA injection (D) TUNEL-positive neurons were not detected in normal brain (E) Immunoblot for BNIP3 demon-strated increased levels of BNIP3 in KA-injected striatum as compared with uninjected CL striatum and normal control rats (Ctrl) Immunopositive blotting for BNIP3 was completely absent when anti-body against BNIP3 was first incubated with

a BNIP3–GST protein (F) Quantification of the western blot bands revealed a nine-fold increase of BNIP3 in KA-injected striatum There was a 3.5-fold increase in CL striatum

of the injected animal as compared with striatum of uninjected animals (G) Levels of BNIP3 mRNA as demonstrated by in situ hybridization were very low in uninjected CL rat striatum (H) Levels of BNIP3 mRNA were increased dramatically following KA injections Scale bars: (A) 500 lm;

(B) 50 lm; (C, D) 200 lm; (G, H) 40 lm.

**P < 0.01.

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domain (Fig 3B), whereas the full-length BNIP3

showed a pattern of punctate localization (Fig 3A)

Neuronal survival rates after 5 days of transfection with

BNIP3 plasmid were decreased (P = 0.0165, n = 3) as

compared with cells transfected with pcDNA3–

hBNIP3)163or the pcDNA3 plasmids (Fig 3C) Among

neurons expressing full-length BNIP3, 62% showed

DNA condensation In contrast, DNA damage was

observed in only 27% of BNIP3-positive neurons

trans-fected with pcDNA3–hBNIP3)163

To demonstrate the role of BNIP3 in glutamate

neu-rotoxicity, we tested the effects of inhibiting BNIP3

expression by RNA interference Hippocampal

neu-rons were infected on day 1 in vitro with the viral

vec-tor pLenti–BNIP3shRNAN167, designed to express a

short hairpin sequence that would target

nucleo-tides 167–188 in the BNIP3 mRNA The vector has

been described elsewhere [18], with an inhibitory effi-ciency of at least 98% for BNIP3 On day 8 in vitro, the neurons were exposed to 100 lm glutamate for

48 h, and cell survival rates were measured with a 3-(4,5-dimethylthiazol-2-yl)-2,5-diphenyl-tetrazolium bro-mide assay As shown in Fig 3D, inhibition of BNIP3 expression increased neuronal survival by 40% (P = 0.0038, n = 4)

To determine whether the BNIP3-mediated cell death pathway in excitotoxicity involved protein syn-thesis, we evaluated the effectiveness of the RNA synthesis inhibitor actinomycin D (1 lgÆmL)1) in pre-venting excitatory neuronal cell death As shown in Fig 4A, addition of actinomycin D decreased cell death caused by glutamate toxicity by 42% (P < 0.01), whereas actinomycin D alone did not affect cell death rates

F

C

0

20

40 60 80

6 12 24 36 48

Hours

Glu100 μ M

Glu10 μ M

D

B

A

Glu 0h 6h 12h 24h 36h 48h 60h 72h Ctrl BNIP3

β-actin

E

Glu (100 μM)

0 2 4 6 8 10

**

*

24 h

12 h

6 h

Fig 2 Glutamate increased BNIP3

expression (A) Glutamate increased

neuro-nal cell death in a dose-dependent manner.

(B) Expression of BNIP3 was not detectable

immunohistochemically in the majority of

untreated neurons; less than 15% of

untreated neurons expressed low levels of

BNIP3 (C) More than 50% of cells treated

with 100 l M glutamate for 6 h stained

posi-tively for BNIP3 (D) Nuclei in BNIP3-positive

neurons showed a dysmorphic appearance

atypical of apoptosis (E) Time course of

BNIP3 expression in neurons exposed to

100 l M glutamate.**P < 0.01; *P < 0.05;

n = 4.

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We next examined caspase involvement in

BNIP3-mediated neuronal cell death Primary hippocampal

neurons were preincubated with z-VAD-FMK (50 lm)

alone or in combination with BOC-D-FMK (50 lm); both of these are potent cell-permeable caspase inhi-bitors Cell viability was determined by trypan blue exclusion 6 h after application of glutamate or N-methyl-d-aspartate (NMDA) NMDA and glutamate significantly increased neuronal cell death (P < 0.01) z-VAD-FMK alone did not prevent cell death caused

by glutamate or NMDA Coapplication of z-VAD-FMK and BOC-D-z-VAD-FMK resulted in a small (17%) but statistically significant decrease in glutamate-induced cell death (P = 0.045, n = 5; Fig 4B)

Discussion

Previously, it was shown for non-neuronal cells that BNIP3 induced cell death distinct from necrosis and apoptosis as defined by classical morphological and molecular criteria [17] It was also shown that excito-toxicity activates cell death programs that result in atypical neuronal cell death [1,5,19] However, at present, it is not completely clear whether and which molecular regulators might control such atypical neu-ronal cell death Accordingly, we tested hypotheses that BNIP3 was an important regulator of neuronal cell death induced by excitotoxic stimuli, that this form of programmed cell death occurred indepen-dently of caspase activation, and that excitotoxic cell death could be prevented if the actions of BNIP3 were blocked Here, we showed that BNIP3 levels increased dramatically in in vivo and in vitro models

of excitotoxicity, that overexpression of full-length BNIP3 decreased the viability of hippocampal neu-rons grown in culture and significantly increased the susceptibility of these neurons to glutamate-induced cell death, that BNIP3-mediated cell death occurred

D

BNIP3-positive cells

with DNA damage

C

0

20

40

60

80

100 **

0 20 40 60 80 100

**

20 μm

20 μm

Fig 3 BNIP3 expression caused neuronal cell death (A) Transient

transfection of rat hippocampal neurons resulted in DNA

condensa-tion and neuronal cell death (B) Transient transfeccondensa-tion of rat

hippo-campal neurons with a dominant-negative form of BNIP3

(BNIP3)163) did not cause DNA condensation or localization of

BNIP3 to mitochondria; BNIP3 was diffusely distributed in the

cyto-plasm (C) Neuronal survival rates after 5 days of transfection with

BNIP3 (n = 6) About 84% of positive neurons in

BNIP3-transfected cells showed DNA condensation, as compared with

27% in BNIP3)163-transfected cells (D) Glutamate significantly

decreased neuronal survival Knockdown of BNIP3 by the lentiviral

vector pLV-N167 significantly protected neurons from

glutamate-induced cell death **P < 0.01, n = 3).

0 20 40 60 80

Media Locke s

Actinom

ycin D Glum

ate

Glu + actinomycin D

0 20 40 60 80

Me

dia

Locke s

Glutam

ate

G + Boc-FMK NMDA Boc-D-FMK Glu +

z-FMK

Glu + Boc-FMK + z-FMK z-VAD-FMK

Fig 4 BNIP3-induced neuronal cell death in excitotoxicity required protein synthesis but was largely independent of caspase activity (A) Actinomycin D significantly decreased the number of trypan blue-positive cells (P < 0.01) caused by glutamate toxicity to untreated control levels (B) Inhibition of caspase activity did not prevent cell death caused by glutamate or NMDA z-VAD-FMK alone did not prevent cell death caused by the excitotoxic toxins Coapplication of z-VAD-FMK and BOC-D-FMK (FMK) resulted in a small but statistically significant decrease in glutamate-induced cell death (P = 0.045).

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independently of caspase activation, and that

inhibi-tion of BNIP3 by RNAi increased neuronal viability

and protected neurons against glutamate-induced

excitotoxicity

BNIP3 is a BH3-only proapoptotic member of the

Bcl-2 family However, unlike in other members of the

Bcl-2 family, the BH3 domain of BNIP3 is not

required for its death-inducing activity Our results

showing that full-length, but not truncated, BNIP3 can

result in neuronal cell death are in agreement with

pre-vious results, obtained with non-neural cells, that the

transmembrane domain of BNIP3 is indispensable for

it to cause membrane damage, mitochondrial

perme-ability, and DNA fragmentation [17] These features of

BNIP3-induced neuronal cell death are

indistinguish-able from those of BNIP3-induced non-neural cell

death [17] BNIP3-regulated cell death appears to be

atypical of necrosis, because it is genetically

pro-grammed (Fig 4) and involves mitochondrial

perme-ability transition pore opening [17] Even though

BNIP3-induced cell death is genetically programmed,

it is atypical of apoptosis because cell death has been

shown to occur independently of caspase activation

and cytochrome c release [17]

BNIP3 expression has been shown to be induced

under conditions of oxidative stress [18] and hypoxia

[20,21] The promoter of the BNIP3 gene contains a

functional hypoxia response element [20] that could be

a direct target of hypoxia-inducible factors

Excitotox-icity involves Ca2+ overloading and concomitant

gen-eration of reactive oxygen species, which has been

shown to trigger hypoxia-induced transcription [22]

Therefore, our studies showing that BNIP3 is both

necessary and sufficient for neuronal death in

excito-toxicity has wide-ranging implications for the

under-standing of mechanisms underlying acute and chronic

neurodegenerative disorders and the possible

identifica-tion of novel therapeutic intervenidentifica-tions

Experimental procedures

Animal model

Male Sprague–Dawley rats, with body weights ranging

between 200 and 250 g, were obtained from the University

of Manitoba Central Animal Care breeding facility All

procedures followed Canadian Council on Animal Care

guidelines and were approved by the Animal Care

Commit-tee at the University of Manitoba Animals were

anesthe-tized with intraperitoneal 74 mgÆkg)1sodium pentobarbital

and placed in a stereotaxic surgery frame Unilateral

intra-striatal injections were performed using the following

coor-dinates (in mm); anteroposterior, 9.0; mediolateral, 3.0; and

dorsoventral, 4.5 [23] Drugs were administered over a

5 min period in a volume of 1 lL, using a 10 lL syringe fit-ted with a 30-gauge needle Following injection, the needle was left in place for 5 min before being slowly withdrawn

to allow diffusion of the drug away from the injection site

KA, dissolved in 50 mm Tris⁄ HCl with the pH adjusted to 7.4 with NaOH, was administered at a dose of 2.5 nmol Control rats received unilateral injections of 1 lL of 50 mm Tris⁄ HCl (pH 7.4) To confirm the role of kainate recep-tors, the receptor antagonist CNQX (dissolved in 0.1 m NaOH with volumes adjusted with 50 mm Tris⁄ HCl,

pH 7.4) was administered at a dose of 5 nmol in a volume

of 1 lL, by itself or in combination with 2.5 nmol of KA Following injection, wounds were sutured, and animals were allowed to recover for periods up to 5 days From pilot studies, we found BNIP3 expression to be increased from 24 h to 5 days after KA injection (data not included)

In the present study, all animals were killed 48 h after intrastriatal injections

Cell culture

Primary hippocampal neurons were prepared from 18-day-old embryonic Sprague–Dawley rats as described previously [24] Briefly, hippocampal tissue was dissociated by gentle tituration in calcium-free Hank’s balanced salt solution, and centrifuged at 1000 g Cells were resuspended in DMEM⁄ F12 nutrient mixture containing 10% heat-inacti-vated fetal bovine serum and 1% antibiotic solution (peni-cillin G 104 IUÆmL)1, streptomycin 10 mgÆmL)1 and amphotericin B 25 lgÆmL)1) in 0.9% NaCl (Sigma, St Louis,

MO, USA) Hippocampal neurons were plated at a density of 2· 105cellsÆmL)1 on 12-mm-diameter poly (d-lysine)-coated glass coverslips Three hours after plating, the medium was replaced with serum-free Neurobasal med-ium containing 1% B-27 supplement (Gibco, Rockville, MD, USA) Immunofluorescent staining for microtubule-associ-ated protein-2 on neurons and glial fibrillary acidic protein

in astrocytes showed that cultures were > 98% neurons; the remainder of the cells were predominantly astrocytes

Pharmacological studies

To determine the extent to which NMDA-type glutamate receptors are involved in BNIP3 expression and excitotoxic cell death, we used the agonist NMDA (100 lm) in the absence or presence of the NMDA receptor antagonist MK-801 (10 lm) To determine the extent to which caspase activation contributes to BNIP3-mediated cell death, hippo-campal cells were incubated in the absence or presence

of the broad-spectrum cell-permeable caspase inhibitors z-VAD-FMK (50 lm) and BOC-D-FMK (50 lm); these inhibitors were applied 30 min prior to application of glutamate or NMDA To determine the role of protein

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translation in glutamate-mediated toxicity, we used

actino-mycin D (1.0 lgÆmL)1)

Plasmids and cell transfection

Rat BNIP3 (rBNIP3) cDNA was prepared by RT-PCR

from primary neuronal cultures exposed to hypoxia for

36 h, with sense primer 5¢-GAGAATTC TCG CAG AGC

GGG GAG GAG AAC-3¢ and antisense primer 5¢-AT

GGATCC TCA AAA GGT ACT AGT GGA AGT TG-3¢ The

PCR product was ligated to pGEM-T (Promega) by T-A

cloning After the resulting construct had been verified

by sequencing, the rBNIP3 fragment was subcloned to

pEGFP-C2(Clontech, USA) to yield green fluorescent

pro-tein–rBNIP3 T7-tagged pcDNA3–hBNIP3 and T7-tagged

pcDNA3–hBNIP3)163 plasmids were gifts from the late A

H Greenberg (University of Manitoba) [14] Transfection

of cells was performed on day 4 in culture with

LipofectA-MINE 2000 (Invitrogen, Burlington, Ontario, Canada),

according to the manufacturer’s protocol The transfection

efficiency was 2–8% as estimated by enhanced green

fluo-rescent protein (EGFP) expression from transfection of

pEGFP-C2–rBNIP3 or by immunohistochemistry with a

monoclonal antibody against T7 (1 : 200; Novagen,

Madi-son, WI, USA) when T7-tagged pcDNA3 plasmids were

used Cells were exposed to glutamate after 9 days in

culture for excitotoxicity experiments

Lentiviral vectors expressing short hairpin RNA

sequences targeting BNIP3 and LacZ have been described

elsewhere [18] Briefly, complementary DNA

oligonucleo-tides targeting rat BNIP3 and LacZ were annealed and

ligated into a pENTR⁄ U6 vector (Invitrogen, San Diego,

CA, USA) The U6 RNAi cassette (U6 promoter +

dou-ble-stranded oligonucleotides + Pol III terminator) was

then transferred to the pLenti6⁄ BLOCK-iT-DEST vector

(Invitrogen) by an LR recombination reaction Lentiviral

stock was produced by transfecting this plasmid into the

293FT Cell Line, using ViraPower Packaging Mix in

DMEM containing 10% fetal bovine serum The lentiviral

stock was titered by counting crystal violet-stained blue

col-onies of 293FT cells after incubation for 3 days with

selec-tive medium containing different concentrations of

blasticidin For transduction, the vectors (multiplicity of

infection = 5) were placed with the neuron in fresh

med-ium 1 day before the neurons were exposed to glutamate

Immunohistochemistry and in situ hybridization

For immunohistochemistry and in situ hybridization, rats

were perfused transcardially with 0.9% saline and then 4%

paraformaldehyde Brains were carefully removed and

post-fixed overnight in NaCl⁄ Pi containing 4%

paraformalde-hyde After being rinsed in NaCl⁄ Pi, the brains were placed

in NaCl⁄ Picontaining 0.5 m sucrose (pH 7.3) at 4C until

buoyancy was lost Eight-micrometer sections were cut on a

cryostat (Shandon) and mounted on silane-treated slides Frozen brain sections cut from KA-injected and control rats were blocked and permeabilized with NaCl⁄ Pi contain-ing 2% BSA, 5% normal goat serum and 0.3% Triton X-100 for 30 min at room temperature The sections were then incubated overnight at 4C with a polyclonal anti-body against BNIP3 (1 : 200), followed by rhodamine-conjugated goat anti-(rabbit IgG) (1 : 200; Jackson ImmunoResearch Laboratories, West Grove, PA, USA) for

2 h at room temperature The polyclonal antibody against BNIP3 recognizes both human and rat BNIP3, and was also used to detect BNIP3 expression in primary rat hippo-campal neurons after exposure to glutamate and NMDA For detection of BNIP3 expression in primary hippocampal neurons after plasmid transfection, a monoclonal antibody against BNIP3 that is specific for human BNIP3 was used

at a dilution of 1 : 200 Fluorescent pictures were taken with a Zeiss (Thornwood, NY, USA) microscope equipped with an AxioCamdigital camera (Carl Zeiss, Jena, Ger-many) For in situ hybridization, an RNA probe (specific for BNIP3) was synthesized with a digoxigenin RNA label-ing kit (Roche) accordlabel-ing to the manufacturer’s protocol Brain sections were hybridized with the probe and incu-bated with an alkaline phosphatase-conjugated antibody against digoxigenin, and labeled cells were detected with BCIP⁄ Nitro Blue tetrazolium

Detection of cell death

In vitro cell death was estimated by trypan blue exclusion Cells were incubated in 0.4% trypan blue solution for

30 min, and then counted under a bright-field microscope Nonviable cells were distinguished by their dark blue stain-ing Neuronal viability was also estimated by 3-(4,5-dim-ethylthiazol-2-yl)-2,5-diphenyl-tetrazolium bromide assay on

a WallacVICTOR31420 Multilabel microplate reader (Per-kin Elmer Life Sciences, Woodbridge, Ontario, Canada) For examination of nuclear morphology, nuclear DNA was stained with Hoescht 33342 DNA fragmentation was detected by TUNEL, using an in situ cell death detection kit with fluorescein (Intergen, Purchase, NY, USA), according

to the manufacturer’s recommendations Morphological characteristics were examined with a Nikon Eclipse TE200 microscope, and fluorescence was examined with a Zeiss Axi-oskop 2 Statistical analyses were perfofmed by ANOVA with Tukey’s post hoc test

Western blots

Rats were killed by decapitation, brains were rapidly removed, and striata were dissected out, frozen rapidly on dry ice, and stored at )80 C For preparation of protein samples, striata were homogenized in 25 mm phosphate buffer (pH 7.4) containing 1% Triton X-100, 0.1 mm EGTA, 1 mm phenylmethanesulfonyl fluoride, and 5 mm

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dithiothreitol After brief centrifugation (1000 g for 10

minutes at 4C), supernatants were collected For cultured

neurons, cell pellets were resuspended in RIPA lysis buffer

(0.01 m Tris⁄ HCl, 0.15 m NaCl, 1% Triton-X 100, 1%

deoxycholic acid, 0.1% SDS, pH 7.4), the lysates were

centrifuged at 1000 g in a microcentrifuge for 10 min

at 4C, and supernatants were collected The protein

con-centration was determined by the Bradford method, with

BSA as standard Protein samples were separated by

SDS⁄ PAGE on a 15% polyacrylamide gel, and transferred

to poly(vinylidene difluoride) membranes suitable for small

molecular mass peptides Proteins were probed with

anti-body against BNIP3 at a dilution of 1 : 500, and

immuno-blotting was detected by electrochemiluminescence

(Amersham, Piscataway, NJ, USA) Controls were run in

the presence of a plasmid-expressed BNIP3 protein

Acknowledgements

This work was supported by the Canadian Institutes of

Health Research, Canadian Stroke Network and the

National Natural Science Foundation of China (Grant

numbers: 81070980⁄ H0910 to ZZ, 30700245 to XX and

U0632007 to TG and JK) J Kong received a New

Investigator award from the Heart and Stroke

Founda-tion of Canada J Weng received a studentship from

the Manitoba Institute of Child Health

References

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