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In vitroproteolysis of DT, using an endosomal lysate, was observed at both neutral and acidic pH, with the subsequent generation of DT-A and DT-B subunits pH 7 or DT fragments with low A

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translocation into the cytosol of rat liver in vivo

Tatiana El Hage1,2, Paulette Decottignies3,4and Franc¸ois Authier1,2

1 INSERM, U756, Chaˆtenay-Malabry, France

2 Universite´ Paris-Sud, Faculte´ de Pharmacie, Chaˆtenay-Malabry, France

3 CNRS, UMR 8619, Orsay, France

4 Universite´ Paris-Sud, Orsay, France

Diphtheria toxin (DT) is the causative agent of the

acute disease diphtheriae and, in mammalian cells,

mediates its cytotoxic effects by catalyzing the transfer

of the ADP-ribosyl moiety of NAD+ to elongation

factor-2 (EF-2), which arrests protein synthesis and kills the cells [1] DT is synthesized by Corynebacte-rium diphtheriaeand belongs to the A–B family of bac-terial exotoxins In the secreted form of DT, the A and

Keywords

cathepsin D; diphtheria toxin; endosome;

furin; translocation

Correspondence

F Authier, Inserm U756, Universite´

Paris-Sud, Faculte´ de Pharmacie, 5 rue

Jean-Baptiste Cle´ment, 92296 Chaˆtenay-Malabry,

France

Fax: +33 1 4683 5844

Tel: +33 1 4683 5528

E-mail: francois.authier@u-psud.fr

(Received 4 July 2007, revised 19 January

2008, accepted 8 February 2008)

doi:10.1111/j.1742-4658.2008.06326.x

A detailed proteolysis study of internalized diphtheria toxin (DT) within rat liver endosomes was undertaken to determine whether DT-resistant species exhibit defects in toxin endocytosis, toxin activation by cellular enzymes or toxin translocation to its cytosolic target Following adminis-tration of a saturating dose of wild-type DT or nontoxic mutant DT (mDT) to rats, rapid endocytosis of the intact 62-kDa toxin was observed coincident with the endosomal association of DT-A (low association) and DT-B (high association) subunits Assessment of the subsequent post-endosomal fate of internalized mDT revealed a sustained endo-lysosomal transfer of the mDT-B subunit accompanied by a net decrease in intact mDT and mDT-A subunit throughout the endo-lysosomal apparatus

In vitroproteolysis of DT, using an endosomal lysate, was observed at both neutral and acidic pH, with the subsequent generation of DT-A and DT-B subunits (pH 7) or DT fragments with low ADP-ribosyltransferase activity (pH 4) Biochemical characterization revealed that the neutral endosomal DT-degrading activity was due to a novel luminal 70-kDa furin enzyme, whereas the aspartic acid protease cathepsin D (EC 3.4.23.5) was identified

as being responsible for toxin degradation at acidic pH Moreover, an absence of in vivo association of the DT-A subunit with cytosolic fractions was identified, as well as an absence of in vitro translocation of the DT-A subunit from cell-free endosomes into the external milieu Based on these findings, we propose that, in rat, resistance to DT may originate from two different mechanisms: the ability of free DT-A subunits to be rapidly pro-teolyzed by acidic cathepsin D within the endosomal lumen, and⁄ or the absence of DT translocation across the endosomal membrane, which may arise from the absence of a functional cytosolic translocation factor previ-ously reported to participate in the export of DT from human endosomes

Abbreviations

CT, cholera toxin; CTF, cytosolic translocation factor; DT, diphtheria toxin; EEA1, early endosome antigen 1; EF-2, elongation factor-2; EN, endosomal fraction; ENs, soluble endosomal extract; HA, hexa- D -arginine; Hsp, heat shock protein; IDE, insulin-degrading enzyme; LPS, postmitochondrial supernatant; PA, pepstatin-A; PE, Pseudomonas exotoxin A; proHB-EGF, heparin-binding epidermal growth factor-like growth factor precursor; TrxR1, thioredoxin reductase-1.

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B moieties are connected by both a peptide bond

(Arg193–Ser194) and a disulfide bridge (Cys186–

Cys201) Proteolytic nicking of the polypeptide and

reduction of the disulfide are required for the A and B

fragments to separate and for cytotoxicity to be

expressed [2]

To access its cytosolic target EF-2, DT must be

transported across cellular membranes and into the

cytoplasm [3,4] The first step in the intoxication

pro-cess involves DT binding, via its B-subunit domain,

to the heparin-binding epidermal growth factor-like

growth factor precursor (proHB-EGF), and it is

believed that DT-DT receptor complexes are

endocyto-sed using a clathrin-dependent pathway [5] Prior to,

during or after endocytosis of DT into early

endo-somes, furin mediates DT cleavage at the consensus

motif within the 14-amino acid loop subtended by the

disulfide bond connecting the A- and B-moieties [6]

After a lag phase of 25 min, two-thirds of the

internal-ized DT is proteolyzed and inactivated within the

endosomal apparatus [7] Three models have been

proposed as being physiologically relevant to the

mechanism of translocation and⁄ or cytosolic release of

the DT catalytic domain: (a) translocation of DT

across the endosomal membrane by its own

transmem-brane domain (tunnel model) [8,9]; (b) translocation of

the DT-A (low association) subunit through the

oligo-meric DT-B (high association) subunit following its

conformational change and insertion into the lipid

bilayer (cleft model) [10]; and (c) the requirement of

chaperonin heat shock protein (Hsp)90 and

thioredox-in reductase-1 (TrxR1) as components of a cytosolic

translocation factor (CTF) [11], as well as cytosolic

factors (ATP, b-COP) [12] Another processing

requirement for internalized DT to become fully active

is reduction of the interchain disulfide bridge, but the

intervening reductive steps, the nature of the relevant

enzymatic activity and the subcellular site at which the

disulfide bond is split (endosome and⁄ or cytosol) are

not understood Nevertheless, reduction of DT may

represent the rate-limiting step in the diphtherial

intox-ication of eukaryotic cells [7]

The existence of cells resistant to DT, such as rodent

cells [13,14], suggests that the above requirements for

DT activation and action may not be present in all

mammalian cells The presence of functional DT

bind-ing sites in rat and mouse cells [15] suggests that the

biochemical determinant(s) for resistance to DT in

rodents must lie distal to the receptor binding step and

involve some aspects of toxin internalization and⁄ or

endosomal degradation and⁄ or translocation of the

DT-A subunit Moreover, novel endosomal fragments

of internalized DT that did not originate from furin

activity have been identified in cells overexpressing DT receptor [16], suggesting that the endosomal proteolytic machinery may also degrade toxins and curtail their action This is in agreement with experiments using furin-deficient cells in which the intracellular inter-action and degradation of internalized DT by unidenti-fied protease(s) was reported [17]

In the present study, we report a detailed processing study of internalized DT within hepatic endosomes obtained from rat, a toxin-resistant species The meth-odology used in the present study, which is the first to use DT and an in vivo model, is similar to that devel-oped to investigate the metabolic fate of internalized cholera toxin (CT) in the endo-lysosomal apparatus [18,19] This methodology has proven useful in relating endosomal processes to toxin cytotoxicity in a physio-logical state Here, we report that rat hepatic endo-somes contain a luminal truncated furin that cleaves the Arg193–Ser194 peptide bond in the connecting A-B region of internalized DT at neutral pH In addi-tion, cathepsin D (EC 3.4.23.5) generates DT cleavages

at the Met14–Glu15 and Leu434–Pro435 peptide bonds of DT at acidic pH and releases DT fragments with low ADP-ribosyltransferase activity These events coincided with an absence of in vivo association of DT

or A with cytosolic fractions isolated from DT-injected rats, as well as an absence of in vitro translo-cation of DT or DT-A from cell-free endosomes into the external milieu

Results

In vivo endocytosis of DT and nontoxic mutant

DT (mDT) within the endo-lysosomal apparatus

of rat liver The kinetics of in vivo uptake of native DT or mDT into the endosomal fraction (EN) were first assessed (Fig 1A) Rats were administered an intravenous injection of either toxin (50 lgÆ100 g)1 body weight) and killed 5–90 min post-injection Hepatic endosomes were isolated and the amount of internalized DT (Fig 1A, upper blots) or mDT (Fig 1A, lower blots) was determined by reducing SDS⁄ PAGE followed by western blotting using rabbit (a-1275) antibody or horse (a-PV) anti-DT serum Following injection of native DT, a short association of the 62-kDa DT form was observed in EN at 5–15 min, whereas the level of individual subunits was maximal at 30 min (DT-A sub-unit) or 30–90 min (DT-B subsub-unit) In response to the

in vivo administration of mDT, which displays a single substitution of Glu52 for Gly in the A-chain, the kinetics of association with hepatic endosomes of

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Fig 1 Kinetics of the appearance of DT and mDT in the endo-lysosomal apparatus after toxin administration (A) EN were isolated at the indicated times after the in vivo administration of native DT (upper blots) or nontoxic mutant mDT (lower blots), and evaluated for their con-tent of internalized toxins by reducing SDS ⁄ PAGE followed by western blot analysis using polyclonal anti-DT 1275 (a-1275; blots on the left)

or PV (a-PV; blots on the right) sera Each lane contained approximately 50 lg of endosomal protein Arrows to the right indicate the mobili-ties of intact toxins ( 62 kDa), B-subunits ( 37 kDa) and A-subunits ( 25 kDa) Molecular mass markers are indicated to the left of each blot (B) The LPS fraction was isolated 15 min after mDT administration, and immediately subfractionated on linear Nycodenz density gradi-ents (panel 4 C), or incubated with ATP and an ATP-regenerating system at 37 C for 60 min prior to subfractionation on linear Nycodenz density gradients (panel 37 C ⁄ 60 min) mDT was evaluated for each subfraction using reducing SDS ⁄ PAGE followed by western blot analy-sis using polyclonal anti-DT 1275 Thirty microliter of each subfraction were loaded onto each lane Arrows to the left indicate the mobilities

of immunodetected mDT ( 62 kDa), mDT-B subunit ( 37 kDa) and mDT-A subunit ( 25 kDa) The LPS fraction was also isolated from control rats and incubated with ATP and an ATP-regenerating system at 37 C for 60 min prior to subfractionation on linear Nycodenz density gradients (lower blots a-EEA1 and a-CD) The content of EEA1 and cathepsin D was evaluated by immunoblotting for each subfraction iso-lated from the Nycodenz gradients Arrows to the right indicate the mobilities of EEA1 (180 kDa), procathepsin D precursor (64 kDa) and mature cathepsin D (45 and 31 kDa) Components appearing at densities in the ranges 1.075–1.105 and 1.11–1.14 gÆmL)1were scored as truly endosomal and lysosomal, respectively.

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mDT, as well as mDT-A or mDT-B subunits, were

similar to that of wild-type DT The levels of both

subunits decreased after 30 min, especially when using

anti-DT 1275 serum, suggesting a degradation state of

DT-A and DT-B subunits at this locus However, a

stronger immunoreactivity of endosome-associated

mDT was observed compared to the nonmutant toxin,

which could result from a higher binding property of

mDT with membrane lipids and proteins [20] and⁄ or a

higher level of mDT endocytosis in rat hepatocytes

Next, we used the in situ liver model system for

analysis of endosome–lysosome transfer to determine

the post-endosomal fate of internalized mDT (Fig 1B)

Transfer of mDT, mDT-A and mDT-B subunits from

the endosomal to the lysosomal compartment was

examined by Nycodenz density gradient analysis of

postmitochondrial supernatant (LPS) fractions

pre-pared 15 min after mDT administration When LPS

fractions were incubated at 4C and analyzed by

reducing SDS⁄ PAGE followed by immunoblot analysis

using rabbit a-1275 antibody (upper blot 4C), most

of the intact mDT and mDT subunits appeared in a single broad region of density in the range 1.070– 1.100 gÆmL)1, which mainly coincided with the endo-somal marker early endosome antigen 1 (EEA1) or procathepsin D precursor (lower blots a-EEA1 and a-CD) When the LPS fraction was incubated at 37C for 60 min (Fig 1B, middle blot 37C ⁄ 60 min), a major shift of the mutant toxin to high density frac-tions was observed, which partially coincided with the lysosomal marker mature cathepsin D enzyme (lower blot a-CD) This was accompanied by a corresponding loss of intact mDT and mDT-A and mDT-B subunits from the endosomal position

Proteolytic activation of DT within hepatic endosomes

We examined the ability of hepatic endosomes to degrade native DT (Fig 2A) The luminal and

Fig 2 Effect of pH and assessment of the degradation products generated from native DT by endosomal DT-degrading activity (A) Total (EN;  10 lg) and soluble (ENs;  1 lg) endosomal fractions were incubated with 10 lg native DT at 37 C for the indicated times in

25 m M Hepes buffer (pH 7) or 25 m M citrate-phosphate buffer pH 4 The incubation mixtures were then analyzed by reducing SDS ⁄ PAGE followed by Coomassie Brilliant Blue staining The major degradation products generated at pH 7 (peptide 7a) or pH 4 (peptides 4a, 4b and 4c) were subjected to N-terminal sequence analysis Arrows to the left indicate the mobilities of intact DT ( 62 kDa), DT-B subunit ( 37 kDa) and DT-A subunit ( 25 kDa) Arrows to the right indicate the mobilities of intact DT ( 62 kDa) and uncharacterized DT frag-ments (60, 35 and 12 kDa) (B) Native DT was digested in vitro for 90 min at 37 C with the ENs fraction in buffer pH 4 or 7 containing dith-iothreitol (0.2 M ) The treated DT (10 lg) was then incubated for the indicated times at 30 C with the cytosolic fraction ( 200 lg) in 0.1 M

Hepes buffer (pH 7.4) in the presence of 1 l M [ 32 P]NAD Samples were then subjected to SDS ⁄ PAGE and analyzed by autoradiography The dried gels were exposed to X-ray film at )80 C for 3 days The arrow to the right indicates the mobility of [ 32 P]-labeled EF-2 ( 105 kDa).

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membrane-bound distribution of endosomal

DT-degrading activity was assessed by reducing SDS⁄ PAGE

analysis of DT digestion performed at pH 7 and 4

(Fig 2A) EN degraded native DT efficiently both at

neutral and acidic pH (Fig 2A, upper gels) DT-A and

DT-B subunits were specifically generated at pH 7,

whereas three major DT fragments of 60, 37 and

12 kDa were observed at pH 4 Subfractionation of

hepatic EN into a soluble endosomal lysate (ENs)

revealed a similar pattern of proteolysis (Fig 2A, lower

gels), suggesting that the majority of the neutral and

acidic DT-degrading activity in endosomes is soluble

The cleavage sites in the major metabolites of DT

were determined by N-terminal sequence analysis

Edman degradation of intermediate 7a generated the

Ser-Val-Gly-Ser-Ser-Leu peptide, suggesting that the

major cleavage produced at neutral pH affected

the peptide bond between Arg193–Ser194 At acidic

pH, N-terminal sequence analysis of the major

DT-fragments revealed cleavages between Met14–Glu15

(as demonstrated by the Glu-Asn-Phe-Ser-Ser-Tyr

sequence at the N-terminal of product 4b) and

Leu434-Pro435 (as demonstrated by the

Pro-Thr-Ile-Pro-Gly-Lys sequence at the N-terminal of product

4c) Moreover, intermediate 4a displayed the

N-termi-nal sequence of DT (Gly-Ala-Asp-Asp-Val-Val), sug-gesting that the cleavage is located within the carboxyl-terminal region of the toxin

We next examined whether, under conditions where

DT was processed by ENs, a corresponding change in the toxin cytotoxicity would be observed (Fig 2B) DT was first partially processed by ENs at pH 4 or 7 under reducing conditions, and then incubated at neu-tral pH with cytosolic fraction in the presence of [32P]NAD A rapid and sustained ADP-ribosylation of cytosolic EF-2 was observed following endosomal pro-teolysis of DT at neutral pH (Fig 2B) However, [32P]ADP-ribose incorporation into EF-2 after endoso-mal digestion of DT under acidic conditions was very low, even after 60 min of incubation (Fig 2B)

Catalytic properties of endosomal neutral and acidic DT-degrading activity

We next evaluated the effects of various protease inhibitors on the neutral (Fig 3A) and acidic (Fig 3B) DT-degrading activity by reducing SDS⁄ PAGE analy-sis The proteolytic activity directed against DT at

pH 7 was inhibited by hexa-d-arginine (HA), a com-petitive inhibitor of furin, and the metal-chelating

A B

Fig 3 Effect of protease inhibitors on the proteolysis of native DT by ENs ENs ( 1 lg) was incubated with 1 l M native DT at 37 C for

180 min in 25 m M Hepes buffer pH 7 (A) or 25 m M citrate-phosphate buffer (pH 4) (B) in the absence or presence of 3.5 lgÆmL)1PA, 1 l M

E64, 1 m M EDTA, 1 l M HA, 1 m M phenylmethanesulfonyl fluoride (PMSF), 1% MeOH or 1% Me2SO (upper gels), or in the presence of HA and PA at the indicated concentrations (lower gels) The incubation mixtures were then analyzed by reducing SDS ⁄ PAGE followed by Coo-massie Brilliant Blue staining Arrows to the right in (A) indicate the mobilities of intact DT ( 62 kDa), DT-B subunit ( 37 kDa) and DT-A subunit ( 25 kDa) Arrows in (B) indicate the mobilities of intact DT ( 62 kDa) and uncharacterized DT fragments (60, 35 and 12 kDa).

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agent EDTA (Fig 3A, upper gels) Neutral

DT-degrading activity was inhibited in a dose-dependent

manner by HA (IC50 1 lm) (Fig 3A, lower gel)

At acidic pH, endosomal DT-degrading activity

was strongly inhibited by pepstatin-A (PA)

(IC50< 10)7m), an inhibitor of aspartic acid

prote-ases, and EDTA (Fig 3B)

Inhibition of acidic DT-degrading activity by PA

and its presence in the endosomal lumen as a soluble

form suggested cathepsin D as a likely candidate for

the degrading activity We therefore used well

charac-terized polyclonal antibodies to mature cathepsin D

and its proform [21,22] to deplete cathepsin D from

ENs (Fig 4A, left gel) Quantitative immunoprecipita-tion of cathepsin D using antibodies directed against the mouse (a-CD R291) or human enzyme (a-CD M8147) removed a major part of the endosomal pro-teolytic activity directed towards DT at pH 4, as assessed by reducing SDS⁄ PAGE analysis Immuno-depletion of ENs with antibodies to cathepsin B and its proform (a-CB 7183) [23] failed to remove the pro-teolytic activity

Hepatic endosomes are known to contain neutral peptidases such as insulin-degrading enzyme (IDE) [24] and furin [25] Endosomal neutral DT-degrading activ-ity was depleted by anti-furin R2 serum directed

A B

C

Fig 4 Effect of immunodepletion of cathepsins and furin on endosomal DT-degrading activity (A) ENs fractions were immunodepleted of active cathepsin D (a-CD), cathepsin B (a-CB), furin (a-furin) or insulin-degrading enzyme (a-IDE) using their respective antibodies, which had been precoated onto protein G-Sepharose beads Following centrifugation, the resultant supernatants were incubated with 1 l M native DT at

37 C for 120 min in 25 m M citrate-phosphate buffer (pH 4) (gel on the left) or 25 m M Hepes buffer (pH 7) (gel on the right) The incubation mixtures were then analyzed by reducing SDS ⁄ PAGE followed by Coomassie Brilliant Blue staining Arrows indicate the mobilities of intact

DT ( 62 kDa) and uncharacterized DT fragments ( 60, 35 and 12 kDa) (B) A total (EN) and soluble (ENs) endosomal fraction, and recombi-nant furin were evaluated by immunoblotting for their immunoreactivity with monoclonal (a-furin R2) or polyclonal (a-furin CT) antibody to furin Arrows indicate the mobilities of intact (90 kDa) or truncated furin (70 and 57 kDa) (C) Rat liver EN were isolated at the indicated times after the in vivo administration of native DT and evaluated by western blotting for their content of furin using monoclonal (a-furin R2) or poly-clonal (a-furin CT) antibody to furin Molecular mass markers are indicated to the left of each blot Arrows to the right indicate the mobilities

of intact (90 kDa) or truncated furin (70 kDa).

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against the catalytic domain of furin (Fig 4A, right

gel) However, immunoprecipitation using antibody to

the carboxyl-terminal domain of furin (a-furin CT) or

to IDE (a-IDE) failed to remove the DT-degrading

activity observed at pH 7

Hepatic endosomes were then evaluated for their

con-tent of furin by immunoblotting with antibodies directed

against either the catalytic or carboxyl-terminal domain

of furin (Fig 4B) The EN showed intense

immuno-reactivity for the 90-kDa furin enzyme using either

anti-furin serum By contrast, anti-anti-furin R2 serum, which is

directed against the catalytic domain of furin, showed

immunoreactivity for the soluble 70-kDa form of furin

in both the total EN and the luminal content (ENs)

Comparably, recombinant truncated furin was revealed

by immunoblotting as a 57-kDa form using anti-furin

R2 serum However, anti-furin CT serum failed to

detect either the 70-kDa soluble form or the 57-kDa

recombinant truncated furin

To determine whether DT alters the degradation

state of furin in the endosomal apparatus, we prepared

endosomes after DT injection and assessed furin

anti-genicity towards both anti-furin sera (Fig 4C) Using

anti-furin R2 serum (left blot), the 90-kDa furin

(major species) was found in endosomes from

untreated rats, as well as the truncated 70-kDa furin

(minor species) Using both anti-furin sera, a

time-dependent decrease in the content of the 90-kDa furin

was observed at 5–60 min after DT injection A

paral-lel increase in the content of the 70-kDa fragment was

observed with anti-furin R2 serum, especially at 5 min

post-injection (Fig 4C, left blot)

To verify the involvement of two enzymes in the

endosomal processing of internalized DT, the

proteo-lytic activity associated with soluble endosomal

pro-teins was further purified on a TSK-GEL G3000

HPLC column (Fig 5) Eluted fractions 5–12 (Fig 5A,

upper panel) were assayed for their content of

cathep-sin D and furin by western blotting ucathep-sing

anti-cathep-sin D R291 or anti-furin R2 sera, respectively

(Fig 5A, blots), as well as for their DT-degrading

activity at pH 7 or 4 using reducing SDS⁄ PAGE

anal-ysis (Fig 5B) The neutral DT-degrading activity

(Fig 5B, fraction 7 of upper gel) coincided with

elu-tion of the 70-kDa immunoreactive furin (Fig 5A,

lower blot) The fraction with the highest acidic

DT-degrading activity (Fig 5B, fraction 9 of lower

gel) revealed a strong immunoreactivity towards the

mature 45-kDa cathepsin D enzyme (Fig 5A, upper

blot) To determine whether furin and cathepsin D are

capable of generating the same degradation products

as those observed with endosomes, DT was subjected

to in vitro digestion with human furin at pH 7

(Fig 5C, upper gel) or bovine cathepsin D at pH 4 (Fig 5C, lower gel) Reducing SDS⁄ PAGE analysis revealed DT proteolytic products with molecular masses identical to those observed with EN or ENs fractions at both neutral and acidic pH (Fig 2A)

Assessment of a functional translocation complex for DT in hepatic endosomes

To evaluate a possible defect in DT-A translocation to its cytosolic target, we measured the presence of the DT-A subunit in cytosolic fractions prepared from DT-injected rats using western blot analysis (Fig 6A)

No detectable immunoreactivity for the DT-A subunit was observed, even at late stages of DT endocytosis (Fig 6A, blot a-DT1275), whereas the cytosolic target

of DT-A, EF-2, was easily detected (Fig 6A, blot a-EF-2) However, a cytosolic translocation of the cytotoxic A-subunit was clearly observed in rats trea-ted with Pseudomonas exotoxin A (PE) (Fig 6A, blot a-PE) or CT (Fig 6A, blot a-CT) at 5–90 min post-injection, consistent with a selective retention of DT-A within rat liver endosomes

To directly assess the translocation of internalized

DT into the extra-endosomal milieu, endosomes iso-lated after injection of DT were incubated in buffered isotonic medium for 30–60 min at 37C in the pres-ence or abspres-ence of ATP, followed by centrifugation at

100 000 g for 60 min (Fig 6B) Western blot analysis

of DT associated with sedimentable endosomes showed

a progressive decrease in immunoreactive DT, as well

as DT-A and DT-B subunits, confirming the process-ing of DT in liver endosomes (Fig 6B, left blot) By contrast, immunoprecipitation of DT from superna-tants followed by western blot analysis using anti-DT

PV serum did not reveal any immunoreactivity towards DT or the individual DT-subunits (Fig 6B, right blot), suggesting that DT and DT degradation products remained strictly associated with endosomes Recently, TrxR1 and the chaperonin Hsp90 have been proposed to be components of a CTF complex that participates in the translocation and release of cytosolic DT-A subunit from early endosomes of human T cells [11] Consequently, we attempted to evaluate the contribution of these enzymes to the endosomal disulfide-reducing activity (TrxR1) and membrane translocation of toxin peptides (TrxR1 and Hsp90) by assessing hepatic endosomes and cytosol for their content of TrxR1 and Hsp90 (Fig 6C) EN iso-lated from control rats (Fig 6C, lane –) revealed a con-centration of TrxR1 equivalent to that observed in the cytosolic fraction (Fig 6C, blots a-TrxR1, lane –)

A weak immunoreactivity for Hsp90 was observed in

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control endosomes (Fig 6C, upper blot a-Hsp90, lane –),

whereas a strong immunoreactivity was detected in

control cytosol (Fig 6C, lower blot Hsp90, lane –) DT

administration did not alter the distribution of TrxR1

or Hsp90 in the endosomal and cytosolic pools

(Fig 6C)

Although these data demonstrate that hepatic

endo-somes may represent a physiological site of TrxR1

localization and action, they do not demonstrate

physi-cal interaction between TrxR1 and internalized DT

Therefore, coimmunoprecipitation studies were

under-taken (Fig 6D) Immunoprecipitation of internalized

DT or TrxR1 from endosomes isolated 15 min after

DT injection was effected with the indicated antibodies followed by immunoblotting with DT PV or

anti-DT 1275 sera As expected, anti-DT, as well as free anti-DT-A and DT-B subunits, were detected in immunoprecipi-tates using anti-DT antibodies However, no DT immunoreactivity was found in TrxR1 immunoprecipi-tates (Fig 6D)

Discussion

The present study aimed to explore the biochemical determinant(s) that confer DT insensitivity to rats

Whereas man, monkey, rabbit, guinea pig and chicken

A B

C

Fig 5 Characterization of endosomal DT-degrading activity by gel-filtration HPLC (A) ENs ( 260 lg) was applied to a TSK-GEL G3000

HPLC column The HPLC profile shows the absorbance at 214 nm The eluted fractions (5–12) were evaluated for their content of cathepsin

D and soluble truncated furin by immunoblotting with their respective polyclonal (a-CD R291) or monoclonal (a-furin R2) antibody Arrows to

the right indicate the mobilities of the immunoreactive proform ( 64 kDa) and mature form ( 45 kDa) of cathepsin D, and soluble

trun-cated furin ( 70 kDa) (B) Eluted fractions (5, 7, 9, 10 and 12) were tested for their ability to degrade 1 l M native DT for 90 min at 37 C in

25 m M Hepes buffer (pH 7) (upper gel) or 25 m M citrate-phosphate buffer (pH 4) (lower gel) The incubation mixtures were then analyzed by

reducing SDS⁄ PAGE followed by Coomassie Brilliant Blue staining Arrows to the right indicate the mobilities of intact DT ( 62 kDa), DT-B

subunit ( 37 kDa), DT-A subunit (25 kDa) and uncharacterized DT fragments (60, 35 and 12 kDa) (C) Native DT (10 lg) was incubated at

37 C with furin (50 UÆmL)1Æmg)1) in 25 m M Hepes buffer (pH 7) containing 6 m M CaCl 2 (upper gel) or cathepsin D (40 UÆmL)1Æmg)1) in

25 m M citrate-phosphate buffer (pH 4) (lower gel) for the indicated times The incubation mixtures were then analyzed by reducing

SDS ⁄ PAGE followed by Coomassie Brilliant Blue staining Arrows to the right indicate the mobilities of intact DT ( 62 kDa), DT-B subunit

( 37 kDa), DT-A subunit ( 25 kDa) and uncharacterized DT fragments (60, 35 and 12 kDa).

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are examples of sensitive species, rats and mice are

examples of insensitive species, requiring a 10 000-fold

higher dose before toxic symptoms are noted, and cell

lines derived from sensitive and insensitive species

exhi-bit a similar range in DT sensitivity [3] However, the

rat has surface membrane receptors for DT that have

properties indistinguishable from the receptors on

sen-sitive cells [4,13] Moreover, rat EF-2 is equally as

good a substrate for in vitro DT-catalyzed

ADP-ribosylation Therefore, the biochemical determinant(s) for DT resistance in rats must lie distal to the receptor binding step and presumably involve some aspects of the endosomal process The present study is consistent with this view Our in vivo and in vitro study supports the contention that the marked degradation of DT-A subunit within the endosomal lumen, as well as the absence of translocation of DT-A subunit to the extra-endosomal milieu (or cytosol), is responsible for part

C D

A B

Fig 6 Assessment of a functional cytosolic translocation complex for DT associated with hepatic endosomes (A) Cytosolic fractions were isolated at the indicated times after the in vivo administration of native DT (blots a-DT1275 and a-EF-2), native PE (blot a-PE) or native CT (blot a-CT), and evaluated for their content of the cytotoxic A-subunit of DT (DT-A), PE (PE-A) or CT (CT-A) using their respective polyclonal antibody, and for their content of EF-2 using polyclonal anti-EF-2 serum Arrows to the left indicate the mobilities of DT-A ( 25 kDa), PE-A ( 37 kDa), CT-A ( 28 kDa) and EF-2 ( 105 kDa) No immunoreactivity for DT-A was observed with cytosolic fractions (B) Membrane translocation of toxin peptides in cell-free endosomes containing in vivo internalized DT The EN was isolated 15 min after the administration

of native DT and then suspended in 0.15 M KCl containing 5 m M MgCl2and, when indicated, 10 m M ATP After the indicated time of incuba-tion at 37 C, endosomes were sedimented by ultracentrifugation and the pellets (endosome-associated material) and supernatants (extra-endosomal material) were evaluated for their content of DT peptides using polyclonal anti-DT 1275 antibody, either directly (pellets) or after

DT immunoprecipitation (supernatants) Arrows to the right and left indicate the mobilities of intact DT ( 62 kDa), DT-B subunit ( 37 kDa) and DT-A subunit ( 25 kDa) No immunoreactivity of DT was observed with supernatants (C) Endosomal and cytosolic fractions were iso-lated at the indicated times after the in vivo administration of native DT, and evaluated by western blotting for their content of TrxR1 and Hsp90 using their respective polyclonal antibody Arrows to the left indicate the mobilities of immunodetected TrxR1 ( 58 kDa) or Hsp90 ( 90 kDa) (D) The EN was isolated 15 min after the in vivo administration of native DT and lysed The endosomal lysate was then immuno-precipitated using anti-DT PV, anti-DT 1275 or anti-TrxR1 sera, and immunoprecipitates were then immunoblotted with antibodies to DT as indicated Arrows to the left indicate the mobilities of DT ( 62 kDa), DT-B subunit ( 37 kDa) and DT-A subunit ( 25 kDa).

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(if not all) of the DT insensitivity in rats Another

pos-sibility is that the rat DT receptor, despite its ability to

bind to and internalize DT, might not be operational

within the endosomal apparatus [14]

The DT receptor concentration of purified rodent

liver plasma membranes is comparable to that found

for the insulin receptor, which is highly expressed on

these same membranes [13] Accordingly, we have

found a marked uptake of DT into rat liver at an early

time of sacrifice (5 min post-injection) after an acute

injection of toxin (50 lgÆ100 g)1 body weight giving a

DT-concentration of approximately 0.25 lm within the

plasma matrix) The present study, which is the first

subcellular fractionation approach to address the

in vivocompartmentalization and processing activation

of DT in rat liver parenchyma, clearly demonstrates

that the toxin is internalized into endosomes as a

sin-gle nonproteolyzed and nonreduced monomeric chain

polypeptide where, at later stages, it undergoes

proteo-lytic processing (major pathway) and reduction (minor

pathway) by the endo-lysosomal apparatus The

kinet-ics of in vivo DT internalization observed in our

bio-chemical studies are comparable to that previously

reported morphologically using in vitro cellular

sys-tems, such as monkey Vero cells [7], human WI-38

cells and mouse 3T3 fibroblasts [26] Indeed, using a

similar in vivo experimental approach to that used in

the present study, we and others have shown that

bac-terial toxins such as CT [18,19] and plant toxins such

as ricin [27] are taken up by the rat liver and

subse-quently accumulate in a low-density endosomal

com-partment where toxin processing begins Our results

extend these observations to DT and show that the

rate of internalization into hepatic endosomes is slower

for CT and ricin (30–40 min) than for DT (5 min)

This may well originate from the ability of CT and

ricin to be endocytosed using both clathrin-dependent

and -independent mechanisms [28], whereas DT

endo-cytosis involves only the clathrin-dependent pathway

[5] Moreover, in the present study, the rate of

endoso-mal processing of internalized DT was much faster

than that of internalized CT [18] This may in part

reflect the two sequential steps in DT processing

(before and after endosomal acidification), whereas

en-dosomal proteolysis of CT involves only the cathepsin

D enzyme and requires endosomal acidification [18,19]

Using the in situ rat liver model system for

endo-some–lysosome fusion [29], we show here, as

previ-ously was the case for native CT [19], a progressive

lysosomal transfer of internalized DT, which was

accompanied by a net decrease in its immunoreactivity

throughout the gradient This result suggests that DT

and DT subunits are proteolyzed within the endosomal

apparatus [16], as well as within lysosomal vesicles The cotransfer of DT subunits to lysosomes in response to DT injection confirms previous studies that have documented a lysosomal association of internal-ized DT in DT sensitive cells [12] and extends this physiological location of DT to the lysosomal appara-tus of insensitive cells

Comparable to the endosomal degradation of inter-nalized ricin A-chain in macrophages [30], the endo-somal processing of internalized DT begins prior to ATP-dependent acidification of the endosomal lumen The endosomal neutral DT-degrading enzyme described in the present study is similar to furin prote-olytic activity in several respects: (a) the neutral DT-degrading activity was inhibited by metal-chelating reagents and hexa-arginine, an inhibitor profile similar

to that of furin [31]; (b) immunoprecipitation of furin from a soluble endosomal extract led to major deple-tion of the neutral degrading activity; and (c) the neu-tral degrading activity produced a cleavage pattern for the DT substrate at the Arg193–Ser194 peptide bond that was similar to that generated using pure furin This is in agreement with previous studies using cellu-lar and acellucellu-lar in vitro systems [6,17] However, the previously reported transmembrane property of furin [31] would argue against a role for furin in the proteo-lysis of DT by a soluble endosomal lysate Interest-ingly, in the present study, we also provide the first evidence for endosomal compartmentalization of a 70-kDa soluble form of furin in rat liver Hence, the 70-kDa furin protein identified is most likely responsi-ble for the endosomal neutral DT-degrading activity due to: (a) its association with soluble endosomal proteins; (b) the similar elution profile of the neutral DT-degrading activity and the immunoreactive 70-kDa furin polypeptide by gel-filtration HPLC; and (c) its binding to monoclonal antibody R2 under nondenatu-rating and denatunondenatu-rating conditions, confirming that the 70-kDa truncated soluble furin represents a proteo-lytically active species

In agreement with our demonstration of a role for a novel soluble 70-kDa furin in the endosomal process-ing of endocytosed DT, the endoprotease furin has been also proposed to catalyze these reactions towards various bacterial toxins both at the cell surface and⁄ or within endocytic compartments [32] Bacterial toxins that require proteolytic cleavage mediated by furin to express full toxic activity include Clostridium septicum alpha-toxin [33], Aerolysin [34], anthrax toxin protec-tive antigen, PE, Shiga and Shiga-like toxins, and bot-ulinum toxin [32]

Immunoblotting in the present study confirmed the detection of furin in rat liver endosomes [25] and rat

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