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Báo cáo khoa học: Glutathione transferases kappa 1 and kappa 2 localize in peroxisomes and mitochondria, respectively, and are involved in lipid metabolism and respiration in Caenorhabditis elegans pot

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peroxisomes and mitochondria, respectively, and areinvolved in lipid metabolism and respiration in Caenorhabditis elegans Elise Petit1,2,3, Xavier Michelet4,5, Claudine Rauch1,2,3, Justi

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peroxisomes and mitochondria, respectively, and are

involved in lipid metabolism and respiration in

Caenorhabditis elegans

Elise Petit1,2,3, Xavier Michelet4,5, Claudine Rauch1,2,3, Justine Bertrand-Michel6, Franc¸ois Terce´6,7,8, Renaud Legouis4,5and Fabrice Morel1,2,3

1 Inserm U620, Universite´ de Rennes 1, France

2 EA-MDC, Universite´ de Rennes 1, France

3 IFR 140, Rennes, France

4 CNRS Centre de Ge´ne´tique Mole´culaire – UPR2167, Gif-sur-Yvette, France

5 Universite´ Paris-Sud Orsay, Universite´ Paris-6, France

6 IFR150, Institut Fe´de´ratif de Recherche Bio-Me´dicale de Toulouse, Plateau technique de Lipidomique, France

7 INSERM, U563, Toulouse, France

8 Universite´ Toulouse III Paul Sabatier, De´partement Lipoprote´ines et Me´diateurs Lipidiques, IFR150, France

Keywords

Caenorhabditis elegans; fatty acids;

glutathione transferase kappa; mitochondria;

peroxisomes

Correspondence

F Morel, INSERM U522 ⁄ EA MDC, Hoˆpital

Pontchaillou, 35033 Rennes, France

Fax: +33 299540137

Tel: +33 299543737

E-mail: fabrice.morel@inserm.fr

R Legouis, Centre de Ge´ne´tique

Mole´culaire – UPR2167 CNRS Baˆtiment 26,

Avenue de la terrasse, 91198 Gif-sur-Yvette

Cedex, France

Tel: +33 169824374

Fax: +33 169824386

E-mail: legouis@cgm.cnrs-gif.fr

(Received 28 May 2009, revised 3 July

2009, accepted 7 July 2009)

doi:10.1111/j.1742-4658.2009.07200.x

To elucidate the function of kappa class glutathione transferases (GSTs) in multicellular organisms, their expression and silencing were investigated in Caenorhabditis elegans In contrast with most vertebrates, which possess only one GST kappa gene, two distinct genes encoding GSTK-1 and GSTK-2 are present in the C elegans genome The amino acid sequences

of GSTK-1 and GSTK-2 share around 30% similarity with the human hGSTK1 sequence and, like the human transferase, GSTK-1 contains a C-terminal peroxisomal targeting sequence gstk-1 and gstk-2 genes show distinct developmental and tissue expression patterns We show that GSTK-2 is localized in the mitochondria and expressed mainly in the phar-ynx, muscles and epidermis, whereas GSTK-1 is restricted to peroxisomes and expressed in the intestine, body wall muscles and epidermis In order

to determine the potential role(s) of GST kappa genes in C elegans, specific silencing of the gstk-1 and gstk-2 genes was performed by an RNA inter-ference approach Knockdown of gstk-1 or gstk-2 had no apparent effect

on C elegans reproduction, development, locomotion or lifespan By con-trast, when biological functions (oxygen consumption and lipid meta-bolism) related to peroxisomes and⁄ or mitochondria were investigated, we observed a significant decrease in respiration rate and a lower concentra-tion of the monounsaturated fatty acid cis-vaccenic acid (18:1x7) when worms were fed on bacteria expressing RNA interference targeting both gstk-1and gstk-2 These results demonstrate that GST kappa, although not essential for the worm’s life, may be involved in energetic and lipid meta-bolism, two functions related to mitochondria and peroxisomes

Abbreviations

Dsba, protein disulfide isomerase A; FAME, fatty acid methyl ester; GFP, green fluorescent protein; GST, glutathione transferase; PTS1, peroxisomal targeting signal 1; RNAi, RNA interference.

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Glutathione transferase (GST) kappa is a 26.5 kDa

protein that was initially isolated from the rat liver

mitochondrial matrix and classified as a class theta

GST [1] The determination of the three-dimensional

structure of the class kappa enzyme from rat

(rGSTK1-1), complexed with glutathione (GSH),

showed a different folding topology from that of the

other GST classes, and revealed that the enzyme shows

similarity with the protein disulfide bond isomerase,

DsbA, from Escherichia coli and a bacterial

2-hydroxy-chromene-2-carboxylate isomerase, an enzyme involved

in the naphthalene degradation pathway [2,3]

Although class kappa GST showed an activity towards

aryl halides, such as 1-chloro-2,4-dinitrobenzene, and

can reduce cumene hydroperoxide and

(S)-15-hydro-peroxy-5,8,11,13-eicosatetraenoic acid [4], this activity

remained quite low when compared with that of other

soluble GSTs Interestingly, a recent study has shown

that GST kappa might also possess a function

inde-pendent of its glutathione conjugation activity in

adi-pose tissue [5] Indeed, Liu et al [5] have identified

GSTK1 as a key regulator for the multimerization of

adiponectin, which is an adipocyte-derived hormone,

in both human and rodent

Tissue distribution, analysed by RT-qPCR, showed

that the hGSTK1 gene is expressed in the 24 different

human tissues examined [4] In the mouse, the

mGSTK1 protein is present in large amounts in the

liver, kidney, stomach and heart, and its association

with liver and kidney mitochondria has been

demon-strated by electron microscopy [6] GSTK1 transcript

tissue expression is similar in the rat and in the mouse

[7] With regard to subcellular localization, in contrast

with soluble GSTs, which are mainly present in the

cytosol, GST kappa is localized in peroxisomes and

mitochondria [4] Although the process of GST kappa

targeting to mitochondria is unclear, it has been

reported to associate with the Hsp60 chaperone [3],

and a possible cleavage site for a mitochondrial

pre-sequence exists at the N-terminus A peroxisomal

tar-geting sequence (tripeptide ARL) has been identified in

the C-terminus of the hGSTK1 subunit [4]

The recent demonstration of GST kappa as a

regu-lator of protein multimerization and its particular

sub-cellular location have led to questions about its further

role(s) and substrate(s) [5] Common peroxisomal and

mitochondrial functions are related to lipid

metabo-lism, including a- and b-oxidation of fatty acids that

generate acetyl-CoA and different acyl-CoA

intermedi-ates [8,9] Thus, the presence of GSTK1 in both

organ-elles suggests that it may be specifically involved in the

b-oxidation of fatty acids, either through its catalytic activity, a certain transport function or interaction with other proteins Interestingly, its role in adipo-nectin regulation is also related to lipid and glucose metabolism

The nematode Caenorhabditis elegans is a genetically well-characterized model organism [10] which presents several advantages: (a) small size; (b) rapid reproduc-tion as a self-fertile hermaphrodite; (c) large number of offspring (250–300 progeny); (d) growth on a solid sur-face medium; and (e) transparent body allowing the observation of cells in mature and developing animals Furthermore, as about 60% of C elegans genes show similarity to human genes, and transient RNA interfer-ence (RNAi) allows specific gene silencing, this model organism represents a powerful tool for gene function analysis

The aim of our study was to characterize GST kappa gene(s) and proteins in C elegans and, by means of RNAi, to investigate the effects of gene silencing on the nematode phenotype Our results showed that the C elegans genome contains two GST kappa genes encoding GSTK-1 and GSTK-2, which localize in peroxisomes and mitochondria, respectively Double inactivation by RNAi affects the worm’s metabolism through a reduction in its rate of respiration and modification of its lipid content

Results

The C elegans genome contains two GST kappa genes

We have previously described a C elegans protein showing 33% homology with the human GST kappa, hGSTK1, amino acid sequence [4] Database analyses revealed the presence of two genes previously named ZK1320.1 and D2024.7 in the C elegans genome These two genes are located on chromosomes II (ZK1320.1) and IV (D2024.7) and have probably arisen by gene duplication Both genes are composed

of three exons and two introns (Fig 1A), the nucleo-tide sequence at the splice junctions is consistent with the canonical GT–AG rule and the corresponding encoded amino acid sequences comprise 226 and 225 residues, respectively, and share 32% identity Ortho-logues of these genes are observed in other nematode species, including C briggsae, C remanei, C japonica, Ancylostoma ceylanicum, Heterorhabditis bacteriophora and Meloidogyne Three arguments strongly suggest

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that C elegans ZK1320.1 and D2024.7 genes belong to

the kappa class of GSTs and share a common

ances-tral gene with rat GSTK1 Firstly, there are conserved

amino acids in the protein sequences of rat GSTK1

and C elegans ZK1320.1 and D2024.7 Secondly, we

showed common exon–exon junctions in the translated

amino acid sequences of the two C elegans genes

and the rat GST kappa gene (Fig 1B) Finally, using

the psortii program (http://psort.ims.u-tokyo.ac.jp),

the presence of a C-terminal peroxisomal targeting

signal 1 (PTS1), composed of the three amino acids

serine-lysine-leucine (SKL), was demonstrated in some,

but not all, nematode species and rat GST kappa

(tripeptide ARL) amino acid sequences (Fig 1C)

Furthermore, psortii also predicted a mitochondrial

presequence with a putative cleavage site after residue

5 (MPNRK⁄ VV) at the N-terminus of the GSTK-2

sequence For these reasons, ZK1320.1 and D2024.7

genes were renamed gstk-1 and gstk-2

GSTK-1 and GSTK-2 are localized in peroxisomes and mitochondria, respectively

The analysis of GST kappa transcript levels has shown

a ubiquitous expression in human [4] and mouse [6] tissues In order to determine the spatial and temporal expression patterns of gstk-1 and gstk-2 genes in

C elegans, we constructed gfp::gstk-1 and gstk-2::gfp fusions under the control of approximately 1 kb of the 5¢ regulatory gstk-1 and gstk-2 sequences, respectively

In these reporter fusion proteins, green fluorescent protein (GFP) was inserted in frame immediately upstream or downstream of gstk-1 and gstk-2 sequences, respectively Transgenic strains were obtained by microinjection, and the localization of fusion proteins in animals was revealed by the exami-nation of GFP fluorescence (Fig 2)

The expression of the gfp::gstk-1 transgene was first detected in 100 cell embryos, as shown in Fig 2A

gstk-1 (zk1320.1) 1 2 3

gstk-2 (d2024.7)

rGSTK1

GSTK-1

GSTK-2

A

B

C

Fig 1 Genomic structure and intron posi-tions in C elegans gst kappa genes (A) Genomic structure of hGSTK1 Exons are represented as black boxes and introns are represented by lines; the numbers indicate the size in nucleotides The gene structure is drawn to scale (B) Intron positions in rat and C elegans amino acid sequences Filled and open triangles mark common and unique intron positions, respectively (C) Amino acid sequence alignment of rat GSTK1 with GST kappa of nematodes The aligned sequences are listed below, followed by the species’ names and accession numbers in parenthe-ses rGSTK1 (Rattus norvegicus, UniProt: P24473), CelGSTK-1 (Caenorhabditis ele-gans, UniProt: Q09652), CbrGSTK-1 (Caenor-habditis briggsae, UniProt: A8XB52), CelGSTK-2 (Caenorhabditis elegans, UniProt: Q18973), CbrGSTK-2 (Caenorhabditis brigg-sae, UniProt: A8X1K2), CreGSTK-2 (Caenor-habditis remanei, WormBase: RP16274), CjaGSTK-1 (Caenorhabditis japonica, WormBase: JA07681), AcGSTK (Ancylos-toma ceylanicum, GenBank: CB175111.1), MhGSTK (Meloidogyne hapla, GenBank: EX007447.1), HbGSTK (Heterorhabditis bacteriophora, GenBank: BM883827.1).

*Residues involved in glutathione binding site.#Residues involved in dimer interface.

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Expression was increased during morphogenesis of the

embryo Fluorescence was observed as strong punctate

staining in intestinal cells and in the epidermis The

number and intensity of fluorescent structures

increased strongly in the intestine during larval

develop-ment (Fig 2D), whereas the epidermal fluorescent

punctata weakened (Fig 2B) In addition, a diffuse

localization of GFP::GSTK-1 was observed in the

body wall muscles (Fig 2C) and in the rectal gland

cells in larvae and adults (Fig 2D) The presence of a

peroxisomal targeting signal in its C-terminus (Fig 1)

and the punctate localization pattern suggest that

GSTK-1 is a peroxisomal protein To confirm the

per-oxisomal localization, we masked PTS1 by fusing the

GFP at the C-terminus of GSTK-1 Transgenic worms

for GSTK-1::GFP only presented diffuse staining (data

not shown), further supporting a peroxisomal

localiza-tion of GSTK-1

In the GSTK-2::GFP transgenic strain, fluorescence

was first detected during the second half of

embryo-genesis (Fig 2E) A strong signal was detected as

punctate staining in the pharynx (Fig 2G) and the

body wall muscles (Fig 2F), and a weaker signal was

also observed in the intestine In muscle cells, the strong GSTK-2::GFP punctata were part of a tubular network which was weakly fluorescent (Fig 2H) Inter-estingly, similar staining has been observed previously

by the expression of GFP fused to specific subcellular targeting sequences [11], strongly suggesting the presence of GSTK-2 in the mitochondria To confirm this mitochondrial localization, we stained both GFP::GSTK-1- and GSTK-2::GFP-expressing worms with MitoTracker Red (Fig 3) Although GFP:: GSTK-1 did not show any colocalization with Mito-Tracker Red staining (Fig 3A¢¢), GSTK-2::GFP fully colocalized with the mitochondrial dye (Fig 3B¢¢) Together, these data indicate that GSTK-1 and GSTK-2 have a peroxisomal and mitochondrial locali-zation, respectively

Impairment of oxygen consumption and lipid content in gstk-1 and gstk-2 double-knockdown worms

Post-transcriptional gene silencing of specific genes by RNAi is a well-established method in C elegans [12]

A

B

C

G F

Fig 2 Analysis of expression pattern of gstk-1 and gstk-2 in embryo and adult C elegans Projection of confocal images of GFP::GSTK-1 (A–D) and GSTK-2::GFP (E–H) at different developmental stages (A) GFP::GSTK-1 is first detected at mid-embryogenesis in the primordium

of the intestine (white arrow) and in the epidermis (arrowheads) A¢ is the corresponding Nomarski picture (B) During larval development, GFP::GSTK-1 is very strongly expressed with a vesicular localization in the intestine (arrows) (C) A weaker expression of GFP::GSTK-1 is present in the muscles (arrowheads) and the epidermis (arrows) (D) Faint diffuse expression is detected in the rectal gland cells (compare with intestinal signal indicated with an arrow) (E) GSTK-2::GFP is first detected in muscle quadrants (arrowheads) during morphogenesis of the embryo E¢ is the corresponding Nomarski picture (F) In larvae, a strong punctate staining is present in the pharynx (arrows) and the body wall muscles (arrowheads), and a weaker signal is observed in the intestine (G) In the pharynx, a very regular expression of GSTK-2::GFP in myo-epithelial cells (arrow) is characteristic of a mitochondrial localization (H) In body wall muscle cells, GSTK-GSTK-2::GFP is detected

as a tubular network with stronger punctata (arrows) typical of the mitochondrial system Scale bar, 10 lm.

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In our study, C elegans (strain N2) was fed with

bac-teria producing dsRNA of the gstk-1 and⁄ or gstk-2

coding regions In each experiment, four feeding

condi-tions were defined: one group of worms was fed with

control bacteria containing the empty plasmid pL4440,

one with bacteria expressing gstk-1(RNAi), another

with bacteria expressing gstk-2(RNAi) and one with a

mix (1 : 1) of both RNAi-expressing bacteria To test

the efficiency of RNAi, fluorescence levels in

RNAi-treated GSTK::GFP transgenic worms were compared

with the levels observed in control worms Figure S1

(see Supporting information) shows that RNAi

directed against either gstk-1 (Fig S1A) or gstk-2

(Fig S1E) was efficient, with the exception of the

pharynx in gstk-2::gfp transgenic worms, where

silenc-ing was incomplete (Fig S1E,F) It is noteworthy that

gstk-2(RNAi) had no effect on the transgenic strain

expressing GFP::GSTK-1 (Fig S1B), and

GSTK-2::GFP expression remained unchanged in transgenic

animals fed with bacteria producing gstk-1 dsRNA

(Fig S1D), indicating the specificity of these RNAi

forms and the absence of compensatory adaptation

[i.e upregulation of gstk-1 in gstk-2(RNAi) worms]

RNAi silencing of gstk-1 or gstk-2 had no apparent

effect on C elegans reproduction or development (data

not shown) This absence of obvious phenotype has

been reported previously in wide RNAi screens (http://

www.wormbase.org) We also found that these RNAi forms did not affect the C elegans lifespan (Fig 4) The first animal died after 5 days and all animals were

Fig 3 GSTK-2, but not GSTK-1, localizes in mitochondria Single confocal images of GFP::GSTK-1 (A) and GSTK-2::GFP (B) in adult animals GSTK-2::GFP fully colocalizes with the mitochondrial-specific marker MitoTracker (A¢), whereas GFP::GSTK-1 is not localized in the mitochondrial network (B¢) Scale bar, 10 lm.

60 70 80 90

100 Control

gstk-1 (RNAi) gstk-2 (RNAi) gstk-1/k-2 (RNAi)

10 20 30 40 50

0

Days

Fig 4 gstk-1(RNAi) and gstk-2(RNAi) do not affect the C elegans lifespan Effects of RNAi-mediated knockdown of gstk-1 and ⁄ or gstk-2 on the lifespan in wild-type worms Worms were fed either with control bacteria not expressing any dsRNA or bacteria expressing dsRNA that targets gstk-1, gstk-2 or both gstk-1 and gstk-2 Nematode survival was analysed by the Kaplan–Meier method using Graphpad Prism 5 The same software was used to test the equality of survival with the log-rank (Wilcoxon) test Each experimental condition was tested in triplicate.

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dead after 32 days The mean lifespan for wild-type,

gstk-1(RNAi), gstk-2(RNAi) and gstk-1+gstk-2

(RNAi) were 14, 14, 13 and 13 days, respectively

These results indicate that gst kappa genes are not

essential for the survival of C elegans

As our expression data for GSTK-1 and GSTK-2

supported peroxisomal and mitochondrial

localiza-tions, we further investigated cellular funclocaliza-tions, such

as lipid metabolism and oxygen consumption, which

are closely related to these two organelles The control

worms showed an oxygen consumption of 12.2 nmolÆ

min)1 per 1000 worms Interestingly, a significant

decrease in oxygen consumption of about 40% (7.5 nmolÆmin)1 per 1000 worms) was observed when worms were fed on bacteria expressing dsRNAs target-ing both gstk-1 and gstk-2 (Fig 5) However, no significant decrease in oxygen consumption was observed in worms fed with 1(RNAi) or gstk-2(RNAi) alone compared with the control condition Thereafter, in order to investigate the effect of gstk-1 and⁄ or gstk-2 silencing on lipid metabolism, we measured the following lipid fractions: phospholipids, diglycerides, triglycerides, free or esterified cholesterol, free fatty acids and total fatty acids Although most lipid concentrations were unchanged (Tables S1–S4, see Supporting information) between wild-type and gstk-1(RNAi), gstk-2(RNAi) and gstk-1⁄ gstk-2(RNAi) worms, a difference was observed for cis-vaccenic acid (18:1x7) for the fatty acid methyl ester (FAME) frac-tion, which was decreased significantly in worms depleted for both gstk-1 and gstk-2 (Fig 6) These data strongly suggest that GSTK-1 and GSTK-2 have overlapping functions, as oxygen consumption and cis-vaccenic acid levels were unchanged in gstk-1(RNAi) and gstk-2(RNAi) worms and decreased only

in double-knockdown animals

Discussion

In this study, we investigated the localization and potential role(s) of GST kappa in C elegans In contrast with most vertebrates, the C elegans genome contains two GST kappa genes, gstk-1 and gstk-2 The two genes are located on different chromosomes and contain three exons Interestingly, orthologues of

C elegans gstk-1 and gstk-2 genes are also found in

0

5

10

15

Control gstk-1

(RNAi)

gstk-2 (RNAi)

gstk-1/k-2 (RNAi)

O 2

RNAi Strain

Fig 5 gstk-1 ⁄ gstk-2(RNAi) animals present an altered respiratory

rate Oxygen consumption was assessed in the fourth larval stage

of animals fed either with control bacteria not expressing any

dsRNA or bacteria expressing dsRNA that targets gstk-1, gstk-2

or both gstk-1 and gstk-2 Results are the mean of six

val-ues ± standard deviation, and are expressed as nmoles of O 2 per

minute per 1000 worms Student’s t-test was applied for statistical

studies between RNAi-fed worms and control worms (*P £ 0.05).

*

Control gstk-1(RNAi) gstk-2(RNAi) gstk-1/k-2(RNAi)

*

8

10

12

14

16

18

Fatty acid mono esters

0

2

4

6

16:0 18:0 16:1w7 18:1w9 18:1w7 18:2w6 20:5w3 17 Cyclo

Fig 6 gstk-1 ⁄ gstk-2(RNAi) animals display

an abnormal FAME composition Simplified

FAME composition of control and gstk-1,

gstk-2 and double gstk-1(RNAi) and

gstk-2(RNAi)-treated animals (see also

Tables S1–S4) Depletion of both gstk-1 and

gstk-2 leads to a decrease in the 18:1w7

fatty acid, but does not affect other lipids.

The results are the mean of three

experi-ments ± standard deviation Student’s t-test

was applied for statistical studies between

RNAi-fed worms and control worms

(*P £ 0.05).

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the nematode species C briggsae and C remanei,

indi-cating that a gene duplication took place before the

speciation of these three Caenorhabditis species

Sequence conservation between the GSTs of C elegans

gstk-1⁄ 2 and those of other nematode species

(C briggsae, C remanei, C japonica, Ancylostoma

ceylanicum, Heterorhabditis bacteriophora and

Meloido-gyne hapla), as well as with rat GSTK1, is also

observed, the most highly conserved residues being

those that contribute to the glutathione-binding site

and dimerization of the protein Interestingly, structure

prediction and molecular modelling studies have

shown that, despite the low sequence similarity

(30%), rGSTK1 and hGSTK1 structures are

recog-nized as the closest structural homologues of C

ele-gans GSTK-1 and GSTK-2 (data not shown)

Together, these observations suggest that C elegans

GSTK-1 and GSTK-2 might have similar activities to

their mammalian orthologues [3,4,7], and may

contrib-ute, at least in part, to detoxification processes

Moreover, a tripeptide sequence (SKL), known as

PTS1, is present at the C-terminal end of the C elegans

GSTK-1 protein PTS1 is involved in protein import

into peroxisomes, and the importance of this signal for

peroxisome targeting in C elegans has been shown

pre-viously by Motley et al [13] Interestingly, GFP fused

to the N-terminus of GSTK-1 was found to localize in

punctate bodies of C elegans cells in several tissues,

strongly suggesting a peroxisomal localization By

con-trast, colabelling with MitoTracker Red showed that

GSTK-2::GFP was present mainly in the mitochondria

It is noteworthy that, in human cells, hGSTK1 is both

peroxisomal and mitochondrial and contains a

C-termi-nal PTS1 [4] This preserved intracellular localization

of GST kappa of both nematodes and vertebrates,

together with sequence conservation and intron

posi-tions in amino acid sequences, indicate that kappa class

genes probably originated from a common ancestral

gene which was present before the protostome⁄

deutero-stome split Interestingly, the presence of two

dupli-cated paralogous genes, gstk-1 and gstk-2, in the

C elegans genome allowed specialization of the

sub-cellular localization for each gene

The use of reporter fusion proteins allowed the

study of tissue expression patterns of gst kappa genes

Although both GFP::GSTK-1 and GSTK-2::GFP

fusion proteins are observed in common tissues, such

as the intestine, they also have a specific localization in

other tissues, such as the epidermis or pharynx for

GFP::GSTK-1 and GSTK-2::GFP, respectively In

C elegans, the intestine and epidermis are at the

interface between the organism and its environment

Therefore, these tissues represent defence barriers

against toxic agents, such as gut-derived oxidants or endogenously generated reactive oxygen species The intestine is also a highly metabolically active organ and represents the tissue in which most C elegans peroxisomes are found, as shown by immunostaining for catalase [14] and by electron microscopy [15] Inter-estingly, GSTK2::GFP is predominantly expressed in muscle cells (pharynx and body wall muscle) The expression of GST kappa genes in body wall muscle and pharynx might be related to the large number of mitochondria in these two tissues, which are associated with high energy consumption

In order to gain further insight into the potential function(s) of GSTK-1 and GSTK-2, RNAi was used

to knock down the expression of the two correspond-ing genes, either separately or simultaneously Knock-down of gstk-1 and⁄ or gstk-2 had no effect on worm lifespan, locomotion or development, suggesting that GST kappa genes are not essential for the worm’s life Because, as in mammals, peroxisomes and mitochon-dria in C elegans play a key role in the production of reactive oxygen species and in lipid metabolism, including fatty acid b-oxidation [16], we investigated the potential role of gstk-1 and⁄ or gstk-2 on the lipid composition of worms For this purpose, phospho-lipids, diglycerides, triglycerides, free and esterified cholesterol, and free and total fatty acid levels were measured in worms fed on gstk-1(RNAi) and⁄ or gstk-2(RNAi) With the exception of cis-vaccenic acid (18:1x7) from the FAME fraction, there was no modi-fication of lipid composition between worms fed on the empty vector control RNAi and those fed on gstk-1(RNAi) and⁄ or gstk-2(RNAi) It is noteworthy that the concentration of cis-vaccenic acid methyl ester was decreased only in double-knockdown (gstk-1 and gstk-2) worms Vaccenic acid is the most abundant fatty acid in phospholipids and triglycerides [17], and

is elongated from palmitoleic acid (16:1x7)

Another phenotypic feature of double-knockdown (gstk-1 and gstk-2) worms was the impairment of oxy-gen consumption It is also noteworthy that vaccenic acid synthesis and worm respiration are closely linked

to peroxisomal and⁄ or mitochondrial activities Inter-estingly, vaccenic acid is an important component of cardiolipin in different animal species [18], and this phospholipid plays a key role in mitochondrial func-tion, particularly at the respiratory chain level [19] Although the link between decreased vaccenic acid levels and impairment in oxygen consumption merits further investigation, the presence of altered phenotypes only in double-knockdown worms indicates compensa-tory roles for GSTK-1 and GSTK-2 and suggests over-lapping functions As peroxisomes and mitochondria

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are metabolically linked, cooperate and cross-talk,

especially in the b-oxidation of various fatty acids and

in the maintenance of homeostasis in cellular reactive

oxygen species [20,21], our results further strengthen

the close relationship between these two organelles It

is noteworthy that specific knockdown of either gstk-1

or gstk-2 was not accompanied by an upregulation of

the paralogous gene Such compensatory responses

have been demonstrated in Gst alpha 4 (Gsta4) or Gst

zeta 1 (Gstz1) knockout mice, where the expression of

other Gst classes and antioxidant enzymes was induced

[22,23] By contrast, knockout of Gst pi1⁄ 2 (Gstp1 ⁄ 2)

did not lead to the upregulation of at least class alpha

and mu transferases, as reported by Henderson et al

[24] As the C elegans genome contains more than 50

genes encoding zeta, sigma, pi and omega class GSTs

[25], further studies are needed to determine whether

GSTs belonging to these classes or other genes are

upregulated in gstk-1 and⁄ or gstk-2 knockout worms

With regard to the potential role of GST kappa,

either direct or indirect, in lipid metabolism and

respi-ration, it stills remain unclear One hypothesis might

be that GST kappa plays a role in the folding of

pro-teins involved in lipid synthesis or respiration Indeed,

it has been demonstrated that GST kappa shares

sequence and secondary structure homology with

E coli protein disulfide bond isomerase (DsbA) and

has the same general folding as DsbA The DsbA

fam-ily is a subfamfam-ily of the thioredioxin famfam-ily and

cata-lyses disulfide bond formation during the folding of

secreted proteins in bacterials [26] Recently, Liu et al

[5] have shown that mouse and human GST kappa,

renamed DsbA-L by these authors, are highly

expressed in adipose tissue and interact with

adiponec-tin Adiponectin is an adipokine specifically secreted

from adipose tissue, which plays a key role in glucose

and lipid metabolism in insulin-sensitive tissues [27]

Overexpression of GST kappa promotes adiponectin

multimerization by the formation of disulfide bonds

between trimers [5] Although there is no adiponectin

gene in the C elegans genome, GST kappa might have

conserved such a role in the regulation of protein

multi-merization or interaction Certain proteins involved in

lipid metabolism can exist as both monomers and

dimers, for example the fatty acid synthase complex,

and it has been demonstrated that some desaturases

also form dimers [28,29] Similarly, several

mitochon-drial proteins form disulfide-linked multimeric

com-plexes [30] Thus, a possible role of GST kappa might

be to favour specific protein–protein interactions, and

the absence of such interactions in double-knockdown

(gstk-1 and gstk-2) worms may lead to lipid metabolism

and respiration impairment Further investigation will

be needed to confirm this hypothesis and to determine which proteins might be regulated by GST kappa

In conclusion, this work has allowed the character-ization of two GST kappa genes, gstk-1 and gstk-2, in the C elegans genome The products of these genes are differentially expressed in worm tissues and show dis-tinct subcellular localizations, namely peroxisomal for GSTK-1 and mitochondrial for GSTK-2 Specific repression of each gene has no consequences on the worm phenotype By contrast, double-knockdown (gstk-1 and gstk-2) worms show decreased vaccenic acid levels and lower oxygen consumption when compared with wild-type worms

Materials and methods

Caenorhabditis elegans strains Caenorhabditis elegans cultures were grown and maintained

at 20C using NGM agar plates supplemented with

5 lgÆmL)1of cholesterol [10] The wild-type reference strain Bristol N2 was used All experiments were performed at

20C

Fluorescent-tagged protein constructs and the production of transgenic animals

Reporter gene constructs were obtained by a PCR fusion-based approach [31] Genomic gstk-2 (D2024.7), with 1.8 kb immediately upstream of the start codon, was PCR amplified from wild-type genomic DNA using a Triple-MasterPCR System (Eppendorf, Hamburg, Germany) This product was then coamplified with a 1.8 kb PCR fragment containing the GFP coding sequence and the 3¢ untrans-formed region (UTR) of unc-54 (from plasmid pPD95.75 kindly provided by A Fire) For gstk-1 (ZK1320.1), a 1.1 kb promoter fragment was amplified and fused with the GFP coding sequence and then coamplified with the gstk-1 geno-mic and 3¢ UTR Sequences were checked and the resulting gfp::gstk-1and gstk-2::gfp fragments were microinjected [32]

at 50 ngÆlL)1 into the syncytial gonad of young wild-type adult hermaphrodites, together with 200 ngÆlL)1of the plas-mid pRF4 containing the dominant marker rol-6(su1006) [33] For each construct, at least three independent lines were analysed for expression

Immunofluorescence microscopy Routinely, fluorescence expression patterns and phenotypic analyses were carried out on a Zeiss axioskop 2 plus equipped with Nomarski optics (Zeiss, Le Pecq, France) Confocal stacks of images every 0.3–0.5 lm were captured

on an inverted Leica SP2 confocal microscope (Leica, Rueil-Malmaison, France) Z projections were analysed

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using Image J software and then processed using Adobe

Photoshop To stain mitochondria, animals were

incu-bated for 10 min with 10 lm of MitoTracker Red

(Invitro-gen Molecular Probes, Cergy Pontoise, France), and then

moved to a fresh plate for 2 h Worms were anaesthetized

in either 1 mgÆmL)1levamisole or 10 lm azide

RNAi experiments

RNAi by feeding bacteria was performed using the N2

strain, as described previously [34,35], with the following

modifications The bacterial HT115(DE3) E coli strains

used for feeding experiments were obtained from J

Ahrin-ger (University of Cambridge, UK) (gstk-1, ref:

WBR-NAi00021881) and OpenBiosystems (Fisher Scientific-Open

Biosystems, Illkirch, France) (gstk-2, ref : RCE1182) In

brief, control and RNAi strain cultures were grown for 6 h

in LB medium containing 100 mgÆmL)1 ampicillin, and

then spread onto NGM agar containing isopropyl

thio-b-d-galactoside (1 mm) and carbenicillin (25 lgÆmL)1) For

dou-ble-RNAi treatment, equal concentrations of both strains

were mixed before seeding The next day, animals at the

fourth larval stage were placed onto RNAi plates, grown

for 2 days and then harvested by rinsing with M9 buffer

(0.1 m NaCl, 0.05 m potassium phosphate, pH 6.0) Adults

were allowed to settle, and eggs were recovered from

hermaphrodites by alkaline hypochlorite lysis (5 min at

room temperature in 0.5 m NaOH, 5% hypochlorite) [36]

The eggs were rinsed with M9 buffer and the resulting L1

larvae were transferred the next day to fresh agar plates

containing the different dsRNA conditions

Lifespan assays

First-generation progeny from RNAi and control

condi-tions were picked at the fourth larval stage and transferred

onto fresh RNAi plates The day of the shift was counted

as day 0 in the adult lifespan assay To prevent mixing test

worms with their progeny during the reproduction period,

adult nematodes were transferred daily to fresh plates

Monitoring of lethality was performed every day and

worms were considered to be dead when they failed to

move, either spontaneously or in response to touch, and

showed no pharyngeal pumping Worms that crawled off

the plate were excluded (considered to have escaped) One

hundred worms per condition were used in each lifespan

experiment, conducted in triplicate Nematode survival was

analysed by the Kaplan–Meier method using Graphpad

Prism 5 The same software was used to test the equality of

survival with the log-rank (Wilcoxon) test

Oxygen consumption assays

Oxygen consumption rates were measured using a DW1⁄ AD

Clark-type oxygen electrode (Hansatech, Norfolk, UK)

Young adult worms that were maintained on NGM agar plates covered with the corresponding RNAi bacteria were washed twice and resuspended in 50 lL of M9, and then transferred into the chamber already containing 450 lL of M9 buffer, and respiration was measured at 20C for at least 10 min All washes and measurements were performed

in oxygenated M9 buffer Samples were carefully recovered from the chamber and the number of worms was counted For each condition, the mean rate was calculated from triplicate experiments

Western blotting

To prepare total extracts, worm pellets were resuspended in Laemmli sample buffer, vortexed three times for 15 s after the addition of broken glass beads, and then denatured for

5 min at 100C and separated by 10% SDS–PAGE Proteins were transferred to nitrocellulose membranes (Schleicher & Schuell BioScience, Dassel, Germany) and probed with the mouse anti-GFP IgG1k (Roche Diag-nostics, Meylan, France) Immunoreactive proteins were revealed with a chemiluminescent detection system (Super-Signal Pico Chemiluminescent Substrate; Pierce Inc., Rockford, IL, USA)

Lipid analyses Aliquots of C elegans were crushed in 2 mL of methanol–

5 mm EGTA (2 : 1, v⁄ v) with an Ultra Turax; 100 lL of homogenate were evaporated and the pellet was dissolved

in 0.25 mL of NaOH (0.1 m) overnight for protein mea-surement using the Bio-Rad assay For each analysis, lipids from the homogenate were extracted according to Bligh and Dyer [37] in chloroform–methanol–water (2.5 : 2.5 : 2.1, v⁄ v ⁄ v) in the presence of the internal standards For total fatty acid analysis, lipids from a 200 lL homogenate were extracted and transmethylated with 1 mL

BF3⁄ CH3OH (SUPELCO 10% w⁄ w) for 1 h at 150 C FAMEs were extracted with 2 mL of hexane–1 mL of water The organic phase was evaporated to dryness and dissolved in 20 lL of ethyl acetate One microlitre of FAME was analysed by gas–liquid chromatography [38] on

a 5890 Hewlett Packard system using a Famewax RESTEK fused silica capillary column (30 m· 0.32 mm inside dia-meter, 0.25 mm film thickness) The oven temperature was programmed from 110 to 220C at a rate of 2 CÆmin)1 and the carrier gas was hydrogen (0.5 bar) The injector and detector were maintained at 225 and 245C, respec-tively Finally, 2 lg of glyceryl triheptadecanoate were used

as internal standard

For free fatty acid analysis, 400 lL of homogenate were extracted and dissolved in 1 mL of hexane Free fatty acids were transmethylated in 1 mL of BF3⁄ CH3OH (10% w⁄ w) for 5 min at room temperature and free FAMEs were extracted with 2 mL of hexane–1 mL of water The organic

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phase was evaporated to dryness and dissolved in 10 lL of

ethyl acetate Analysis was performed as above with 1 lg

of nonadecanoic acid as internal standard All chemicals

were obtained from Sigma-Aldrich, Lyon, France

Acknowledgements

This work was supported in part by the Institut

National de la Sante´ et de la Recherche Me´dicale,

Centre National de la Recherche Scientifique and

Association pour la Recherche sur le Cancer Elise

Petit was founded by the Ligue National Contre le

Cancer and Xavier Michelet by the Association pour

la Recherche contre le Cancer We are grateful to

Pro-fessor A Guillouzo and Drs E Culetto and B

From-enty for critical reading of the manuscript The

Imaging and Cell Biology Facility of the IFR87

(FR-W2251) ‘La plante et son environnement’ is

sup-ported by the Action de Soutien a` la Technologie et la

Recherche en Essonne, Conseil de l’Essonne

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