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reversible reaction of mammalian ferrochelataseMasayoshi Sakaino1, Mutsumi Ishigaki1, Yoshiko Ohgari1, Sakihito Kitajima1, Ryuichi Masaki2, Akitsugu Yamamoto3and Shigeru Taketani1,4 1 De

Trang 1

reversible reaction of mammalian ferrochelatase

Masayoshi Sakaino1, Mutsumi Ishigaki1, Yoshiko Ohgari1, Sakihito Kitajima1, Ryuichi Masaki2, Akitsugu Yamamoto3and Shigeru Taketani1,4

1 Department of Biotechnology, Kyoto Institute of Technology, Japan

2 The First Department of Physiology, Kansai Medical University, Moriguchi, Osaka, Japan

3 Faculty of Bioscience, Nagahama Institute of Bioscience and Technology, Nagahama, Shiga, Japan

4 Insect Biomedical Center, Kyoto Institute of Technology, Japan

Keywords

ferrochelatase; inner membrane; iron

removal; mitochondrial outer membrane;

phosphorylation

Correspondence

S Taketani, Department of Biotechnology,

Kyoto Institute of Technology, Sakyo-ku,

Kyoto 606-8585, Japan

Fax: +81 75 724 7789

Tel: +81 75 724 7789

E-mail: taketani@kit.ac.jp

(Received 9 May 2009, revised 18 June

2009, accepted 25 July 2009)

doi:10.1111/j.1742-4658.2009.07248.x

Ferrochelatase catalyzes the insertion of ferrous ions into protoporphyrin

IX to produce heme Previously, it was found that this enzyme also partici-pates in the reverse reaction of iron removal from heme To clarify the role

of the reverse reaction of ferrochelatase in cells, mouse liver mitochondria were fractionated to examine the localization of ferrochelatase, and it was found that the enzyme localizes not only to the inner membrane, but also to the outer membrane Observations by immunoelectron microscopy con-firmed the dual localization of ferrochelatase in ferrochelatase-expressing human embryonic kidney cells and mouse liver mitochondria The conven-tional (zinc-insertion) activities of the enzyme in the inner and outer mem-branes were similar, whereas the iron-removal activity was high in the outer membrane 2D gel analysis revealed that two types of the enzyme with dif-ferent isoelectric points were present in mitochondria, and the acidic form, which was enriched in the outer membrane, was found to be phosphory-lated Mutation of human ferrochelatase showed that serine residues at positions 130 and 303 were phosphorylated, and serine at position 130 may

be involved in the balance of the reversible catalytic reaction When mouse erythroleukemia cells were treated with 12-O-tetradecanoyl-phorbol 13-ace-tate, an activator of protein kinase C, or hemin, phospho-ferrochelatase levels increased, with a concomitant decrease in zinc-insertion activity and a slight increase in iron-removal activity These results suggest that ferrochela-tase localizes to both the mitochondrial outer and inner membranes and that the change in the equilibrium position of the forward and reverse activ-ities may be regulated by the phosphorylation of ferrochelatase

Structured digital abstract

l MINT-7233234 : Ferrochelatase (uniprotkb: P22315 ), Abcb7 (uniprotkb: Q61102 ) and b5 reduc-tase (uniprotkb: Q9DCN2 ) colocalize ( MI:0403 ) by cosedimentation through density gradients ( MI:0029 )

l MINT-7233207 : b5 reductase (uniprotkb: Q9DCN2 ), COXIV (uniprotkb: P19783 ), Abcb7 (uni-protkb: Q61102 ) and Ferrochelatase (uniprotkb: P22315 ) colocalize ( MI:0403 ) by cosedimenta-tion through density gradients ( MI:0029 )

l MINT-7233195 : ATP synthase (uniprotkb: Q50DL5 ) and Ferrochelatase (uniprotkb: P22315 ) colocalize ( MI:0403 ) by fluorescence microscopy ( MI:0416 )

Abbreviations

AIF, apoptosis inducible factor; b 5 -reductase, NADH-cytochrome b 5 reductase; COX IV, cytochrome c oxidase subunit IV; MDH, malate dehydrogenase; MEL, mouse erythroleukemia; PKC, protein kinase C; TPA, 12-O-tetradecanoyl-phorbol 13-acetate.

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In the last step in the heme biosynthetic pathway,

ferrochelatase catalyzes the insertion of ferrous ions

into protoporphyrin IX to form protoheme The

mam-malian enzyme is nuclear encoded, synthesized as a

precursor form (48 kDa), and translocated into the

mitochondrion, where it is proteolytically processed to

its mature size of 41–42 kDa [1,2] The active site of

the mammalian enzyme faces the matrix of the

mito-chondrion [3] The enzyme not only utilizes ferrous

ions as a substrate in vivo, but also inserts divalent

metal ions such as zinc and cobaltic ions into the

por-phyrin ring in vitro [4,5] Thus, the enzyme is able to

synthesize metalloporphyrins in vitro, although the

uti-lization of ferrous ions to form heme in cells is strictly

controlled [6] Recently, the reverse reaction of

ferroch-elatase, namely the removal of iron from heme, was

reported to occur both in vivo and in vitro [7]

Ferroch-elatase in the heme-requiring pathogen Haemophilus

influenzae functions in the reverse reaction, enabling

the bacteria to obtain iron from the host [8] The yeast

and bacterial enzymes also exhibit the reverse reaction,

although the role of the reverse reaction is unclear [7]

Because hemoproteins, including myoglobin and

hemoglobin, become substrates of the removal reaction

of ferrochelatase [7], the question arises as to how the

cytoplasmic protein myoglobin-heme is moved to the

matrix side of the inner membrane of mitochondria,

where ferrochelatase is known to be located

More-over, how the forward and reverse reactions of

ferrochelatase are regulated in cells has not been

dem-onstrated To clarify the utilization of heme for the

reverse reaction of ferrochelatase, the localization of

ferrochelatase in mitochondria was re-examined It was

found that ferrochelatase is localized in the outer and

inner membranes of mitochondria The translation

product of ferrochelatase is a single isoform, and the

presequence corresponding to the mitochondrial

recog-nition signal is present at the N-terminus of the

trans-lation product, resulting in the targeting of the enzyme

to mitochondria Dual localizations of some

mitochon-drial proteins have been reported previously [9,10],

although the mechanisms involved in the differential

localization of the same translational product have not

been demonstrated

The phosphorylation of various mitochondrial

pro-teins has been established [11] The presence of protein

kinases in the inner membrane of mitochondria may

play a role in the modulation of mitochondrial

func-tions in various tissues For example, some subunits of

cytochrome oxidase are phosphorylated both in vivo

and in vitro [12] NADH dehydrogenase and pyruvate

dehydrogenase are phosphorylated and their activities are changed for physiological purposes [13,14] Although ferrochelatase activity is modulated by lipids and heavy metal ions [1,15], the post-translational modification of ferrochelatase to address these differ-ent functions has not been reported The presdiffer-ent study reports the localization of ferrochelatase in the outer and inner membranes of mitochondria and the possible regulation of its reversible enzyme activity by phos-phorylation Phosphorylation of the enzyme may relate

to the activities and differential localization of ferr-ochelatase A new recycling pathway of heme that includes the iron-removal reaction of heme at the sur-face of mitochondria is proposed

Results

Localization of ferrochelatase in mitochondria

To examine the localization of ferrochelatase, the cDNA for ferrochelatase was transfected into Cos-7 cells and the localization of expressed ferrochelatase was compared with that of an inner membrane pro-tein, ATP synthase Immunofluorescence analysis with anti-ferrochelatase sera indicated that mouse ferroch-elatase appeared predominantly in mitochondria, which was similar to the location of ATP synthase in Cos-7 cells (Fig 1A) Mitochondria and cytosol from mouse liver were fractionated and the conventional zinc-chelating (forward) and iron-removal (reverse) activities, corresponding to the two ferrochelatase activities (Fig 1B), were examined in the mitochondria and cytosol The activity of cytochrome c oxidase, an inner membrane protein, was only found in the mito-chondrial fraction, whereas the activity of malate dehy-drogenase (MDH), a matrix enzyme of mitochondria, was mostly found in mitochondria, although approxi-mately 15% of the activity leaked into the cytosol Large parts of the zinc-chelating and iron-removal activities were found in mitochondria, and approxi-mately 10% of both enzyme activities were in the cytosol Although ferrochelatase is known to be a membrane-bound protein [1,2], these results suggested that some ferrochelatase had leaked to the cytosolic fraction To examine how the enzyme is bound to the mitochondrial membrane, mitochondria were frozen and thawed, and then separated from the supernatants (Fig 1C) Immunoblot analysis showed that ferrochela-tase was found in the supernatants, indicating that ferr-ochelatase is a peripheral membrane protein, as revealed

by the deduced amino acid sequence of mammalian

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ferrochelatase [1,16] Next, mitochondria were purified

from the crude mitochondrial fraction, and intact

mitochondria were treated with trypsin or Na2CO3 As

shown in Fig 2A, an immunoblot analysis revealed

that the amount of NADH-cytochrome b5 reductase

(b5-reductase), a protein located in the outer

mem-brane, was markedly decreased by trypsin treatment,

whereas inner membrane proteins cytochrome c

oxi-dase subunit IV (COX IV) and ABCB7 remained

unchanged, indicating that the surface of outer

mem-brane was digested by trypsin The amount of

ferroch-elatase in the mitochondria was decreased by trypsin

treatment, suggesting that a part of the ferrochelatase

protein is located at the surface of mitochondria

Alka-line (0.1 m Na2CO3) treatment of mitochondria

mark-edly reduced the level of ferrochelatase, demonstrating

that the enzyme at the surface and inside of

mitochon-dria is bound peripherally to membranes To examine

the location of ferrochelatase in detail, purified

mito-chondria were fractionated into the outer and inner

membrane fractions (Fig 2B) Compared with the

proteins located in the outer and inner membranes,

ferrochelatase was located in both membranes of

mouse liver mitochondria, and approximately 60% of ferrochelatase was found in the inner membrane The iron-removal activity in the outer membrane was higher than that in the inner membrane, whereas the forward (zinc-chelating) activity was similar in both membranes (Fig 2C–E)

Electron microscopic analysis of ferrochelatase localization

To further examine the localization of ferrochelatase, pcDNA-HA-FECH was transfected into human embryonic kidney HEK293T cells, after which the cells were fixed, and cryo-ultrathin sections of the cells were processed for immunogold labeling Gold particles (10 nm) showed the presence of HA-tag ferrochelatase bound to the outer membrane of mitochondria and co-localized with TOM 20 (5 nm gold particles), as well as to the inner side of mitochondria around the inner membrane (Fig 3A) The location of ferrochela-tase was confirmed by co-localization with an inner membrane protein, apoptosis inducible factor (AIF) (Fig 3B) When immunostaining by anti-ferrochelatase

Merged

0 0.2 0.4 0.6 0.8

1 1.2

Mitochondria Cytosol

MDH Cytochrome oxidase Zinc insertion Iron removal

1

Supernatants

Pellets

43

43

kDa-2 3

A B

C

Fig 1 (A) Mitochondrial localization Cos-7 cells were transfected with pcDNA-HA-FECH, and incubated for 23 h They were then fixed, per-meabilized and reacted simultaneously with anti-ATP synthase and anti-HA sera to demonstrate localization of ATP synthase and ferrochela-tase The merged exposure confirms that the dots co-localize Scale bar = 10 lm (B) Subcellular distribution of the ferrochelatase activity of mouse liver After mouse liver was homogenized, the cell debris and nuclear fraction were removed Mitochondria were separated by centri-fugation and washed Cytosol was obtained from the post-mitochondrial supernatant by centricentri-fugation at 105 000 g for 60 min Ferrochela-tase activities, including zinc-insertion and iron-removal reactions, were measured The activities of MDH and cytochrome c oxidase were also measured The values are the average of three independent experiments (C) The release of ferrochelatase from mitochondria Isolated miotchondria were untreated (lane 1), frozen at )30 C and thawed twice (lane 2), and the freeze-thawed treatment was repeated (lane 3) The treated mitochondria were separated from supernatants by centrifugation at 105 000 g for 60 min Aliquots were withdrawn and immu-noblotting was performed with anti-ferrochelatase serum.

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sera was performed using cryo-ultrathin sections of

mouse liver, ferrochelatase was detected in both the

outer membrane and the inner part of mitochondria

(Fig 3C) These observations indicated that

ferrochela-tase was localized not only in the inner membrane, but

also in the outer membrane of mitochondria

Phosphorylation of ferrochelatase

Next, we examined whether the different locations of

ferrochelatase lead to different structural and

func-tional properties Ferrochelatase from the inner and

outer membranes was analyzed by 2D gel

electropho-resis (Fig 4A) Immunoblot analysis revealed that the

IEF point of ferrochelatase of size 42 kDa was

differ-ent between the inner and outer membrane forms

Namely, the protein from the outer membrane was

more acidic than that from the inner membrane

Anti-phosphoserine sera reacted with acidic ferrochelatase

(Fig 4B) Immunoprecitated ferrochelatase (HA-tag)

from human embryonic kidney cells expressing

HA-ferrochelatase was phosphorylated (Fig 4C) When the

phosphorylation of ferrochelatase was compared

between the inner and outer membranes, ferrochelatase

in the outer membrane was more heavily

phosphory-lated than that in the inner membrane (Fig 4D) Pre-viously, ferrochelatase was separately purified by the conventional iron-insertion activity using Blue-Sepha-rose [4,15] and iron-removal activity using Red-Aga-rose [7], and 2D gel analysis of the purified enzyme showed the ferrochelatase bound to blue dye was more basic than that bound to red-dye (Fig 4E) When comparing the peptides from these two ferrochelatase enzymes by MALDI-TOF MS, three tryptic peptides containing serine residues at positions 130, 303 and

330 were found to be different These serine residues were conserved among yeast, bacteria and mammalian enzymes It is possible that these serine residues can be phosphorylated Therefore, three mutated ferrochelata-ses were constructed, expressed and purified from Esc-herichia coli When ferrochelatase was phosphorylated

in E coli, (Fig 4F, lower), the intensity of phospho-ferrochelatase of S130A and S303A was decreased, and the band was not detected in the double mutant S130A and S303A, indicating that ferrochelatase was phosphorylated at positions 130 and 303 The reaction

of ferrochelatase with anti-phosphoserine sera was unchanged by the S330A mutation, indicating that ser-ine at position 330 is not phosphorylated When the conventional zinc-chelating activity in these mutants

Ferrochelatase

COXIV

ABCB7

B5-reductase

1.6

0.2

0.6

1

0

1.2

Relative protein levels None Trypsin Na

2 CO 3

0 0.2 0.4 0.6 0.8 1 1.2

Whole Inner membrane

Outer membrane

Ferrochelatase COXIV ABCB7 B5-reductase

Ferrochelatase

COXIV

ABCB7

B5-reductase

None Trypsin Na 2 CO 3

Mitochondria

Whole Inner membrane Outer membrane

Ferrochelatase

COXIV

ABCB7

membrane

Outer membrane

–1 protein·h

–1 )

50

0

100

Zn-mesoporphyrin formed (nmol·mg

–1 protein·h

–1 )

0 100 200 300 400 500 600 700

A

Fig 2 Submitochondrial location of ferrochelatase (A) Trypsin or alkaline treatment Purified mitochondria were treated with trypsin (150 lgÆmL)1) or 0.1 M Na2CO3for 30 min on ice Immunoblotting was carried out with antibodies for ferrochelatase, Cox IV, ABCB7 and b5 reductase (B) Densitometric quantitation of mitochondrial proteins Values are expressed as the mean ± SD of four experiments (C) Loca-tion of ferrochelatase in the outer and inner membranes Mitochondria were separated into inner and outer membranes and immunoblotting was performed (D) Densitometric quantitation of ferrochelatase, COX IV, ABCB7 and b5-reductase of the inner and outer membranes (E) Ferrochelatase activity in outer and inner membranes Zinc-insertion and iron-removal activities were measured in the outer and inner membranes The values obtained are the mean ± SD of three experiments.

Trang 5

was examined, S130A and S330A decreased to 20%

and 68% of wild-type, respectively, and S303A did not

show any activity (Fig 4F, upper) The iron-removal

activity of S130A was similar to that of control, but

that of S330A was 55% of the control No activity

was observed in S303A These results suggest that

ser-ine at position 303 is essential for the catalytic activity

and that phosphorylation of serine at position 130

may be involved in the regulation of the forward

reac-tion of ferrochelatase

An increase in the acidic form of ferrochelatase

in 12-O-tetradecanoyl-phorbol 13-acetate

(TPA)- or hemin-treated mouse erythroleukemia

(MEL) cells

Finally, we attempted to clarify the possible regulation

of phosphorylation of ferrochelatase When MEL cells

are treated with hemin, the cells can utilize

exoge-nously added heme and initiate erythroid

differentia-tion [17,18] Accordingly, cell extracts from 50 lm

hemin-treated MEL cells were analyzed by 2D gel

elec-trophoresis The phosphorylation of ferrochelatase was examined by treatment of the cells with TPA, a typical activator of protein kinase C (PKC), for 6 h, as a posi-tive control As shown in Fig 5A, most ferrochelatase

in TPA-treated cells appeared as a single spot at an acidic site, whereas major two spots were observed in untreated cells MEL cells were then treated with

50 lm hemin and ferrochelatase was analyzed by 2D gel analysis One major spot of ferrochelatase was found at the position of the acidic site (Fig 5A) Phos-phoserine levels corresponding to the position of ferrochelatase increased in hemin-treated cells (data not shown) The forward activity of the enzyme in TPA-treated cells was decreased, whereas the iron-removal activity increased slightly In hemin-treated cells, iron-removal activity also increased, but the insertion of zinc ions into mesoporphyrin decreased (Fig 5B) These results suggest that phosphorylation

of ferrochelatase in MEL cells, as mediated by PKC, led to a decrease of the conventional ferrochelatase activity, indicating a preference for the removal of iron from heme

TOM 20/Ferrochelatase

Ferrochelatase

AIF/Ferrochelatase

A

C

B

Fig 3 Immunoelectron microscopic

analy-ses of the localization of ferrochelatase (A)

HEK293T cells were transfected with

pcDNA-HA-FECH and cryo-ultrathin sections

were double stained by immunogold

meth-ods Anti-HA (10 nm gold particles) and

anti-TOM 20 (arrows, 5 nm gold particles) were

used Scale bars = 0.1 lm (B) Cryo-ultrathin

sections of HEK293T cells, as above, were

labeled with anti-HA (10 nm gold particles)

and anti-AIF (arrows, 5 nm gold particles).

(C) Cryo-ultrathin sections of mouse liver

were labeled with anti-ferrochelatase serum

and 10 nm immunogold particles.

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Lysates Control IgG Anti-HA

HA

Immunoprecipitation

Immunoblots

P- Serine

P- Serine P- Serine

P- Serine

Ferrochelatase

Mitochondria

Outer Membrane

Membrane

Outer membrane

Inner membrane

Outer membrane + inner membrane

10

pH

43 kDa

43 kDa

10 pH

Ferrochelatase

pH

10

kDa

Blue

Red

43-43- Blue + Red

100

50

0

/ S303A S303A S330A S130A

S130A Wt

Ferrochelatase

Zn-mesoporphyrin formed (µ

–1 protein·h

–1 )

Protoporphyrin formed (pmol·mg

–1 protein·h

–1 )

0 0.5 1 1.5 2 2.5 3

C

D

Fig 4 2D gel analysis of ferrochelatase (A) Mitochondria were fractionated into the inner and outer membranes The mitochondrial proteins from both membrane fractions were analyzed by 2D gel electrophoresis Immunoblotting with anti-ferrochelatase serum was performed (B) Mitochondrial proteins were analyzed by 2D gel electrophoresis and immunoblotting was performed with anti-ferrochelatase and anti-phos-phoserine sera (C) HEK293T cells were transfected with pcDNA-HA-FECH and solubilized using 1% Triton X-100 After centrifugation at

15 000 g for 20 min, immunoprecipitation with anti-HA serum was carried out, followed by immunoblotting with ant-HA and anti-phospho-serine sera (D) Mitochondrial proteins from the inner and outer membranes were analyzed by SDS-PAGE and labeled with anti-ferrochela-tase and anti-phosphoserine (E) Ferrochelaanti-ferrochela-tases purified from Blue-Sepharose and Red-Agarose were analyzed by 2D gel electrophoresis Immunoblotting was performed with anti-ferrochelatase serum (F) Wild-type and mutated (S130A, S303A and S330A) ferrochelatases were expressed in E coli Cellular proteins were analyzed and immunoblotting was performed with anti-phosphoserine and anti-ferrochelatase sera (lower panel) The zinc-insertion and iron-removal activities of ferrochelatase were measured (upper panel) Data are the mean ± SD of three independent experiments.

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The present study first demonstrated that mammalian

ferrochelatase is located not only in the inner

mem-brane, but also in the outer membrane of

mitochon-dria Immunoblot data revealed that approximately

60% of the enzyme in mouse liver mitochondria was

present in the inner membrane and the remaining

enzyme with a similar molecular mass was in the outer

membrane Electron microscope observations

con-firmed the outer and inner membrane localization of

ferrochelatase A previous study [7] demonstrated that

the enzyme exhibited two catalytic reactions:

iron-insertion into porphyrin and the removal of iron from

porphyrin The reversible reaction of ferrochelatase

may be ascribed to the different location Because the

myoglobin-heme can be utilized for the removal

reac-tion of iron from heme [7], ferrochelatase located in

the outer membrane of mitochondria is able to contact directly with cytosolic myoglobin Thus, the outer membrane enzyme may demonstrate a preference for the iron-removal reaction b5-Reductase is localized not only in the endoplasmic reticulum, but also the outer membrane of mitochondria in various tissues [19,20], suggesting that the ferric ions of hemoproteins, including myoglobin and hemoglobin, are reduced by this enzyme, and that the reduced ferrous ions can be removed by ferrochelatase

We previously reported [21] that mammalian fer-rochelatase was purified from various tissues using blue dye, but did not bind to red dye Conversely, the enzyme catalyzing removal of iron from heme was purified using Red-Agarose and identified as ferroch-elatase Analysis of the purified ferrochelatases from red and blue dyes by 2D gel analysis revealed that they exhibited different isoelectric points (Fig 4E), indicat-ing the occurrence of post-translational modification of ferrochelatase Various mitochondrial enzymes, such as cytochrome c oxidase and aconitase, are phosphory-lated, and reversible phosphorylation may play an important role in mitochondrial function [11] The present data clearly showed that one of the phophory-lated proteins is ferrochelatase Considering that fer-rochelatase located in the outer membrane exhibited

an acidic isoelectric point by 2D gel analysis (Fig 4A), the enzyme in the outer membrane is mainly phos-phorylated The newly-synthesized ferrochelatase con-tains a pre-sequence at the N-terminus, which is cleaved during the processing into the inner membrane

of mitochondria [1] Because ferrochelatase in the outer membrane has a molecular mass similar to that

of the enzyme in the inner membrane, the movement

of the enzyme to the outer membrane may occur after the cleavage of the pre-sequence, and may relate to the phosphorylation

Mutation studies with ferrochelatase showed that serine residues at positions 130 and 303 were phos-phorylated (Fig 4F) The zinc-insertion activity of S130A mutant was low compared to that of the wild-type enzyme, whereas the iron-removal activity of the mutant was similar to the wild-type enzyme Further-more, the treatment of mitochondria with alkaline phosphatase resulted in a decrease in the iron-removal reaction (data not shown) Thus, the phosphorylation

of serine at 130 may contribute to a change in the equilibrium position of the reverse reaction of the enzyme

It has been reported that more than 50 mitochon-drial proteins are phosphorylated [11] The phosphory-lation of these proteins is mediated by various protein kinases, including protein kinase A and PKC [11,22]

10

Untreated

Hemin

TPA

-43 kDa

-43 kDa

-43 kDa

0

40

80

120

160

200

240

280

320

Protoporphyrin formed (pmol·mg

–1 protein·h

–1 )

30

20

10

0

Zn-mesoporphyrin formed (nmol·mg

–1 protein·h

–1 )

TPA

A

B

Fig 5 Phosphorylation of ferrochelatase in hemin- and TPA-treated

MEL cells (A) MEL cells were treated with 50 l M hemin and

10 n M TPA for 6 h The cellular proteins were analyzed by 2D gel

electrophoresis and immunoblotting was performed using

anti-fer-rochelatase serum (B) The zinc-insertion and iron-removal activities

of ferrochelatase with extracts from cells untreated or treated with

hemin and TPA were measured Data are the mean ± SD of three

independent experiments.

Trang 8

The data obtained in the present study indicated that

the phosphorylation of ferrochelatase in MEL cells

was enhanced by treatment with TPA and hemin

Because TPA is known to be an activator of PKC, the

phosphorylation of ferrochelatase may be mainly

medi-ated by PKC Immunoelectron microscopic

observa-tions revealed that the kinase was associated with the

inner membrane and cristae [23], and physiological

studies suggested that PKC isoforms play a direct role

in regulating mitochondrial functions Because the

acti-vation of PKC induces apoptosis [24–26], the

phos-phorylation of mitochondrial proteins by PKC may

lead to the inhibition of mitochondrial functions

Simi-lar to the case for heme biosynthesis, activation of

PKC repressed the expression of d-aminolevulinic

syn-thase-1, with a concomitant increase in expression of

heme oxygenase-1 [27–29] Thus, PKC may be

involved in the decrease in the intracellular level of

heme to help depress mitochondrial functions by

reducing the production of mitochondrial

hemopro-teins The present study demonstrated that the increase

in phosphorylated ferrochelatase in TPA- or

hemin-treated cells caused a decrease in the metal

ions-insertion reaction, indicating that phosphorylated

ferrochelatase functions in the suppression of heme

biosynthesis

Previously, it was demonstrated that the treatment

of MEL cells with hemin for 24–48 h resulted in an

increase in the mRNA and protein levels of

ferrochela-tase [30,31] The ferrochelaferrochela-tase activity in MEL cells

treated with hemin for 2–3 days also increased [1,32]

By contrast to data demonstrating that ferrochelatase

levels increased in hemin-treated MEL cells [18,30], the

data obtained in the present study showed that

treat-ment of cells with hemin for 6 h resulted in a decrease

in activity Because a short period of treatment of the

cells with hemin caused an increase in the

phosphory-lation of ferrochelatase, with a concomitant decrease

in the zinc-insertion reaction, but not the iron-removal

reaction, phosphorylated ferrochelatase prefers to

remove iron from heme of exogenously added hemin,

suggesting that the iron-removal activity plays a role

in decreasing the level of uncommitted heme in cells

The discrepancy between short- and long-period

treat-ments with hemin has not been explained, although it

is possible that additional regulation may exist in the

expression of ferrochelatase, which plays a role in the

iron-removal reaction of exogenous heme and the

change in position of the heme-moiety of

hemopro-teins The protoporphyrin ring of the heme-moiety in

hemoproteins is re-used and utilized for the new

syn-thesis of hemoproteins after the re-insertion of ferrous

ions This recycling system of protoporphyrin-heme is

markedly induced, accompanied by the induction of

de novo biosynthesis of heme [7] during erythroid differentiation, indicating that this may be necessary for the supply of heme to apo-proteins located in com-partments different from those of the original proteins

Experimental procedures

Materials

Mesoporphyrin IX was purchased from Porphyrin Products (Logan, UT, USA) Restriction endonucleases and DNA modifying enzymes were obtained from Takara Co (Tokyo, Japan) and Toyobo Co (Tokyo, Japan) Antibod-ies for bovine ferrochelatase and b5-reductase (methemoglo-bin reductase) were produced as described previously [4,7] Anti-ATP-synthase and anti-phosphoserine sera were obtained from Millipore-Upstate (Tokyo, Japan) and Zymed Laboratory (San Francisco, CA, USA), respectively Anti-COX IV sera was from Abcom Co (Tokyo, Japan) Anti-TOM 20, anti-AIF and anti-actin sera were products

of Santa Crutz Co (Santa Crutz, CA, USA) Percoll was obtained from Fulka Biochemika (Steinheim, Sweden) Ferrochelatase was purified using Blue-Sepharose (GE Healthcare Biosciences, Amersham, UK) or Red-Agarose (Millipore Corp., Bedford, MA, USA) and the purified enzyme was digested with trypsin, followed by peptide anal-ysis by MALDI-TOF MS [7] All other chemicals were of analytical grade

Plasmids

The full-length cDNA of mouse ferrochelatase [16] was digested with KpnI and ligated into KpnI-digested pcDNA3 (HA) vector [33] The resulting plasmid, pcDNA-HA-FECH, was introduced into E coli XL1-Blue

Cell culture and DNA transfection

Monkey kidney Cos-7 cells, MEL cells and human embry-onic kidney HEK293-T cells were grown in DMEM supple-mented with 10% fetal bovine serum and antibiotics The cells were transfected using Lipofectamine (Invitrogen Co., San Jose, CA, USA) or calcium phosphate with pcDNA-HA-FECH and were then incubated in the presence of fetal bovine serum at 37C for the specified period [34]

Isolation and subfractionation of mouse liver mitochondria

Mouse liver mitochondria were isolated by differential centrifugation [7,15] and purified further by a self-forming Percoll gradient centrifugation according to the method of Hoppel et al [35] To separate the outer membrane from

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the inner membrane, the mitochondria pellet was

resus-pended in 20 mm potassium phosphate⁄ 0.2% defatted

BSA⁄ 1 mm NaVO4 (pH 7.2) (0.2 mg proteinÆmL)1) and

incubated on ice with gentle stirring to induce swelling and

rupture of the mitochondrial outer membrane After

20 min, ATP and MgCl2were added at final concentrations

of 1 mm each, and the suspension was stirred for a further

5 min on ice The swelling⁄ shrunk mitochondria were

cen-trifuged for 20 min at 4C at 22 550 g and the pellet was

gently resuspended in 50 mL of 20 mm potassium

phos-phate⁄ 0.2% defatted BSA ⁄ 1 mm NaVO4 (pH 7.2) The

mitochondrial suspension was treated with two strokes of a

tight-fitting pestle (Wheaton Industries Inc., Millville, NJ,

USA) and centrifuged at 1900 g for 15 min at 4C The

supernatant was removed and centrifuged for 20 min at

22 550 g to obtain the crude outer membrane pellet The

outer membrane was purified by centrifugation at

121 000 g using a discontinuous sucrose gradient consisting

of 17%, 25%, 35% and 60% The 25–35% sucrose fraction

was diluted 10-fold with 20 mm potassium phosphate⁄ 1 mm

NaVO4(pH 7.2) and the pellet was recovered by

centrifuga-tion at 184 000 g To isolate the inner membrane, the

1900 g pellet was loaded onto a discontinuous sucrose

gra-dient consisting of 17%, 25%, 37.5%, 50% and 61% and

centrifugation at 100 000 g at 4C for 16 h [35,36] The

35–40% sucrose fraction was collected and diluted diluted

10-fold with 20 mm potassium phosphate⁄ 1 mm NaVO4

(pH 7.2) The inner membrane was recovered by

centrifuga-tion at 22 550 g at 4C for 1 h The cytosolic fraction was

obtained from the post-mitochondrial supernatant by

centrifugation at 105 000 g at 4C for 60 min to remove

microsomes

Alkaline treatment and trypsin digestion of

mitochondria

Purified mitochondria were treated with trypsin

150 lgÆmL)1 for 30 min on ice and then trypsin inhibitor

(300 lgÆmL)1) was added The trypsin-treated mitochondria

were collected by centrifugation at 9000 g for 10 min To

collect membrane proteins from mitochondria,

mitochon-dria were treated with 0.1 m Na2CO3 for 30 min on ice,

and the membrane fraction was collected by centrifugation

at 9000 g at 4C for 10 min [37]

2D gel analysis

Proteins were first analyzed on the basis of charge by IEF

and then by size, using SDS-PAGE Briefly, mitochondrial

proteins were separated by IEF using an ATTO 2D agar

gel (pH 3.5–10) (ATTO Corp., Tokyo, Japan) IEF ran at

300 V for 150 min After the first-dimension IEF, the tube

was removed from the glass tube and loaded onto a slab

SDS-polyacrylamide gel (10%) for electrophoresis in the

second dimension at 100 V for 2 h

Immunoblotting

Cellular and mitochondrial proteins were separated by SDS-PAGE and transferred to a poly(vinylidene difluoride) membrane (Bio-Rad Laboratories, Hercules, CA, USA) Conditions for immunoblotting for ferrochelatase and other antigens, and the detection of cross-reacted antigens, were performed as described previously [7,34] The relative level

of proteins was quantitated by scanning the band using ATTO Image Freezer AE-6905

Immunofluorescence microscopy

Cos-7 cells were washed with NaCl⁄ Pi(+) (NaCl⁄ Pi contain-ing 1 mm CaCl2and 0.5 mm MgCl2), fixed with 4% parafor-maldehyde for 20 min, and permeabilized in 0.1% Triton X-100 in NaCl⁄ Pi(+) for 1 h After blocking with 2% fetal bovine serum in NaCl⁄ Pi(+), incubation with anti-HA as the primary antibody was carried out, followed by incubation with Cy3-conjugated goat anti-(mouse Ig) (BD Biosciences Co.) [34] For double staining experiments, the cells were fur-ther incubated with anti-ATP synthase (Millipore Co., Tokyo, Japan), followed by Cy2-conjugated goat anti-(rabbit Ig) (Becton-Dickinson Biosciences, Franklin Lakes, NJ, USA) The cells were examined using a Carl Zeiss LSM 510 confocal microscope (Carl Zeiss, Oberkochen, Germany)

Immunoelectron microscopy

Cryo-ultramicrotomy and double-immunogold staining on the cryo-ultrathin sections were performed as described pre-viously [38] with slight modifications Briefly, HEK293T cells were transfected with pcDNA-HA-FECH and the pellet of HEK293T cells was fixed in 4% paraformaldehyde in 0.1 m sodium phosphate buffer (pH 7.4) for 30 min Mouse liver was perfusion-fixed through the heart with 4% paraformal-dehyde in 0.1 m phosphate buffer (pH 7.4) for 10 min Fixed HEK293T cells and liver tissue were processed for ultrathin cryosectioning Frozen sections of HEK293T cells were incu-bated with mixture of monoclonal anti-HA mouse serum and polyclonal anti-TOM 20 or anti-AIF rabbit sera, followed by incubation with a mixture of anti-mouse IgG coupled with

10 nm gold particles and anti-rabbit IgG coupled with 5 nm gold particles Frozen sections of HEK293T cells were incu-bated with polyclonal anti-ferrochelatase rabbit serum and then anti-rabbit IgG coupled with 10 nm gold particles Stained sections were negatively stained, embedded in poly-vinyl alcohol [39], and examined using a Hitachi H7600 electron microscope (Hitachi, Tokyo, Japan)

Enzyme assay

The reaction mixture for iron-removal activity contained

25 mm potassium phosphate buffer (pH 5.7), 50 lm

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hemin-imidazole, 2 mm EDTA, and 2 mm ascorbate, in a final

volume of 1.0 mL in a Thunberg vacuum tube The air in

the tube was replaced with nitrogen gas and dissolved gas

was removed in vacuo [7] The reaction was carried out at

45C for 1 h After the resulting mixture was centrifuged

at 1000 g for 10 min at room temperature, fluorescence was

measured in the supernatant by scanning 550–700 nm

fluo-rescence emissions with excitation at 400 nm The

zinc-chelating ferrochelatase activity was measured as described

previously [7,40] The activities of cytochrome c oxidase

and MDH were measured by the methods of Yamamoto

et al.[41] and Kitto et al [42], respectively

Recombinant enzymes

Human wild-type ferrochelatase protein carrying a his-tag

was described previously [40] cDNAs for mutated human

ferrochelatase S130A, S303A and S330A were prepared: in

the first round of PCR, human ferrochelatase [43] was used

as a template Primer pairs used were primer A (5¢-AAG

AATTCGGTGCAAAACCTCAAGT-3¢) as forward

pri-mer, a mutagenic primer (5¢-GAGGCGGAGCCCCCATC

-3¢), primer B (5¢-AAAAGCTTCACAGCTGCTGGCTGG

-3¢) as reverse primer, and the mutagenic primer (5¢-GATG

GGGGCTCCGCCTC-3¢) for the substitution S130A In

the preparation of the substitution S303A, 5¢-TGTGGC

AAGCCAAGG-3¢ and 5¢-AACCTTGGCTTGCCACA-3¢

were used as mutagenic primers In the case of the

substitu-tion S330A, 5¢-CATTTACCGCTGCCCATA-3¢ and 5¢-TA

TGGGCAGCGGTAAATG-3¢ were used In the second

round, the (A) and (B) primer pair was used to amplify the

full-length human ferrochelatase sequences with the

muta-tion, and the DNA fragments were purified, sequenced and

inserted into a pET vector as described above Plasmids

FECH, FECH S130A, FECH S303A,

pET-FECH S330A and pET-pET-FECH S303A⁄ S330A were

intro-duced into E coli strain BL21 Proteins were overexpressed

and purified as described previously [40]

Acknowledgements

We thank Drs T Ogishima, H Otera and K Mihara

for the kind gifts of anti-b5 reductase and anti-TOM

20, respectively; Dr Y Iwai for the kind gift of

pcDNA3-HA vector; Drs T Endo and T Kataoka for

valuable advice; and S Gotoh and Y Kohno for

providing excellent technical assistance This study was

supported in part by grants from the Ministry of

Education, Science, Sports and Culture of Japan

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