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Exploration of the diaphorase activity of neutrophil NADPH oxidaseCritical assessment of the interaction of iodonitrotetrazolium with the oxidase redox components Alexandra Poinas1, Jacq

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Exploration of the diaphorase activity of neutrophil NADPH oxidase

Critical assessment of the interaction of iodonitrotetrazolium

with the oxidase redox components

Alexandra Poinas1, Jacques Gaillard2, Pierre Vignais1and Jacques Doussiere1

1 Laboratoire de Biochimie et Biophysique des Syste`mes Inte´gre´s, UMR 5092 CEA-CNRS, De´partement de Biologie Mole´culaire

et Structurale Grenoble, France;2De´partement de Recherche Fondamentale sur la Matie´re Condense´e SCIB-SCPM,

CEA-Grenoble, France

In the O2 generating flavocytochrome b, the

membrane-bound component of the neutrophil NADPH oxidase,

electrons are transported from NADPH to O2 in the

fol-lowing sequence: NADPHfi FAD fi heme b fi O2

Although p-iodonitrotetrazolium (INT) has frequently been

used as a probe of the diaphorase activity of the neutrophil

flavocytochrome b, the propensity of its radical to interact

reversibly with O2led us to question its specificity This study

was undertaken to reexamine the interaction of INT with the

redox components of the neutrophil flavocytochrome b

Two series of inhibitors were used, namely the flavin analog

5-deaza FAD and the heme inhibitors bipyridyl and

ben-zylimidazole The following results indicate that INT reacts

preferentially with the hemes rather than with the FAD

redox center of flavocytochrome b and is not therefore a

specific probe of the diaphorase activity of

flavocyto-chrome b First, in anaerobiosis, reduced heme b in activa-ted membranes was reoxidized by INT as efficiently as by O2 even in the presence of concentrations of 5-deaza FAD which fully inhibited the NADPH oxidase activity Second, the titration curve of dithionite-reduced heme b in neutro-phil membranes obtained by oxidation with increasing amounts of INT was strictly superimposable on that of dithionite-reduced hemin Third, INT competitively inhib-ited the O2uptake by the activated NADPH oxidase in a cell-free system Finally, the heme inhibitor bipyridyl com-petitively inhibited the reduction of INT in anaerobiosis, and the oxygen uptake in aerobiosis

Keywords: diaphorase; INT reductase; NADPH oxidase; neutrophils; flavocytochrome b

Upon activation, the neutrophil NADPH oxidase complex

generates the superoxide anion O2 from which are derived

microbicidal oxygen species, such as hydrogen peroxide and

hypochloride The active NADPH oxidase complex consists

of a membrane-bound flavocytochrome b made of two

subunits, gp91phox and p22phox (phox for phagocyte

oxidase), and water-soluble proteins of cytosolic origin

(p67phox, p47phox, p40phox and Rac 1/2) [1] A defect in

any of the genes encoding gp91phox, p22phox, p47phox or

p67phox results in chronic granulomatous disease (CGD)

[2] Physiological activation of NADPH oxidase can be

mimicked by using a cell-free system with flavocytochrome b,

p47phox, p67phox, Rac, GTP and arachidonic acid as basic

components The large subunit of flavocytochrome b,

gp91phox, contains all of the redox components necessary

for electron transfer from NADPH to O2, namely FAD and

two hemes [3–6] Like the yeast FRE1 reductase, the b

cytochrome of the mitochondrial bc1 complex and cyto-chrome b6 of the b6f complex in chloroplasts, gp91phox contains multiple hydrophobic domains, consistent with transmembrane a helices, and two pairs of histidine residues

in these hydrophobic domains, separated by 13 intervening amino acids (quoted from [7]) Based on these considera-tions, it has been postulated that the two hemes located in the N-terminal domain of gp91phox are coordinated by two pairs of histidine residues within two distinct a helices [7] One of them (heme 1) is close to the cytosolic face of the membrane, the other (heme 2) is on the opposite side of the membrane The C-terminal region of gp91phox, which consists of predominantly hydrophilic amino acid residues,

is extramembranous and exposed to the cytosol It contains binding sites for NADPH and FAD The FAD binding site

is thought to be in the close neighborhood of heme 1 The topographical assignment of the redox centers of gp91phox

in this model indicates that electrons are transported from NADPH to O2across the membrane via a chain of redox components in the following sequence: NADPHfi FADfi heme 1 fi heme 2 fi O2 Consistent with the presence of two distinct domains in gp91phox are reports showing that gp91phox may act as a diaphorase in the presence of appropriate electron acceptors such as dichlo-rophenol indophenol [8] or p-iodonitrotetrazolium violet (INT) [9–11] Not only the oxidase activity, but also the diaphorase activity required activation for full elicitation [8,9] From these studies emerged the idea that the electron flux along the redox components of flavocytochrome b is

Correspondence to J Doussiere DBMS/BBSI, CEA-Grenoble,

17 rue des Martyrs, 38054 Grenoble cedex 9, France.

Fax: +33 4 76 88 51 85, Tel.: +33 4 76 88 34 76,

E-mail: jdoussiere@cea.fr

Abbreviations: INT, P-iodonitrotetrazolium; NBT, nitroblue

tetrazolium; 5-deaza FAD, 5 deazaflavin adenine dinucleotide; SOD,

superoxide dismutase; CGD, chronic granulomatous disease; phox,

phagocyte oxidase.

(Received 24 September 2001, revised 30 November 2001, accepted

3 January 2002)

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regulated both at the level of the NADPH-FAD portion of

the electron transfer chain and at the level of heme b [12,13]

This idea was supported by the finding that neutrophils of a

CGD (91X+) patient with a point mutation (Arg54Ser) in

the gp91phox subunit of flavocytochrome b, although

unable to reduce O2into O2 despite the presence of heme b

in gp91phox, retained the capacity to reduce INT [14] It was

also shown that electron transfer from NADPH to INT and

from NADPH to O2 could be activated independently of

each other, depending on the presence of p67phox and

p47phox [10,12] From these data it appeared that

measu-rement of INT reductase could be taken as an index of the

diaphorase activity of flavocytochrome b However, a recent

paper [15] called attention to the possibility of the

nonen-zymatic univalent reduction of tetrazolium salts, particularly

INT by O2 with concomitant production of the tetrazolium

radical In addition, the INT radical in an aerated medium

can reduce O2to O2 [16,17] These observations suggested

that under certain circumstances INT did not probe the

diaphorase activity of the NADPH oxidase The present

paper describes experiments in which INT and O2 were

compared for their ability to accept electrons from activated

flavocytochrome b, using neutrophil membranes pretreated

with 5-deaza FAD, an FAD analog inefficient in electron

transfer in flavocytochrome b, and with benzylimidazole

and bipyridyl as heme inhibitors The results show that INT

is able to directly oxidize reduced heme b

E X P E R I M E N T A L P R O C E D U R E S

Materials

NADPH, ATP, GTPcS were from Boehringer; horse heart

cytochrome c type III, arachidonic acid,

dimethanesulfox-ide, diisopropyl fluorophosphate, benzylimidazole and

hemin were from Sigma INT was from Amresco 5-Deaza

FAD was a gift from V Massey, Medical School Ann

Harbor Michigan (USA)

Biological preparations

Neutrophil membranes and cytosol were prepared from

bovine neutrophils in saline phosphate buffer (NaCl/Pi)

composed of 2.7 mM KCl, 136.7 mM NaCl, 1.5 mM

KH2PO4 and 8.1 mM Na2HPO4, pH 7.4 supplemented

with 1 mMdiisopropyl fluorophosphate and 1 mMEDTA

[13] Protein concentration was assayed with the BCA

reagent using BSA as standard Purified flavocytochrome b

in detergent was obtained as reported previously [18]

Preparation of INT radical

INT (60 mg) was solubilized in 1 mL of a mixture of

dimethyl sulfoxide/H2O (2 : 1, v/v) The oxidized INT was

reduced by a few grains of sodium dithionite The pale

yellow solution became rapidly orange, which is typical of

the INT radical This was immediately followed by four

sequential extractions of INT by 2 mL chloroform After

each extraction, the mixture was centrifuged at 1000 g for

2 min The chloroform solutions containing INT were

collected and pooled After evaporation under a flow of

nitrogen, the dry residue (57 mg) was taken up in 2 mL

dimethyl sulfoxide and kept at)20 °C under argon

Assay of oxidase and INT diaphorase activities NADPH oxidase activity was assayed in a cell-free system [13], either by measurement of the rate of production of O2

or the rate of O2 uptake INT diaphorase activity was assayed by the rate of reduction of INT into formazan in the presence of superoxide dismutase (SOD) or under anaero-biosis In all cases, the assay of oxidase activity was preceded

by an activation step at room temperature Briefly, mem-branes obtained from resting neutrophils were mixed with

2 mMMgSO4and an optimal amount of arachidonic acid After 5 min, cytosol from resting cells (an amount corres-ponding to 10· that of membrane protein), 20 lMGTPcS,

500 lMATP and 2 mMMgSO4were added, and incubation was continued for another 5 min In the case of O2 measurement, 10–20 lg aliquots of membrane protein were used in a final volume of 20–50 lL of NaCl/Pi Following activation, the suspension was transferred to a photometric cuvette containing 200 lM NADPH and either 100 lM cytochrome c or 100 lM INT in 2 mL NaCl/Pi Cyto-chrome c reduction was recorded at 550 nm (e¼ 21.1 mM )1Æcm)1) [19], and INT reduction at 500 nm (e¼ 11 mM )1Æ1 cm)1) [20] After 2–3 min, 50 lg of SOD was added to quench O2 In all preparations, cytochrome c reduction was inhibited to > 95% by the addition of SOD, indicating that O2 was the main product of reduction of O2

In contrast to the reduction of cytochrome c which is monoelectronic, reduction of INT to formazan requires two electrons For normalization of the data, the activities were calculated as lmol e–transferredÆmin)1Æmg membrane pro-tein)1 When reduction of INT was conducted under anaerobiosis, the cuvette containing the medium was sealed with gas-tight rubber stoppers, into which two needles were inserted One of the needles was used for flushing nitrogen, the other for gas evacuation

When the oxidase activity was assayed by the rate of O2 uptake, the suspension of activated particles was transferred

to an oxygraphic cuvette containing 1.5 mL NaCl/Pi supplemented with 250 lMNADPH and, when indicated, INT or heme inhibitors at different concentrations The quantity of neutrophil membranes used in the oxygraphic assays was 10· that used in the photometric assay For measurement of the Kmof activated oxidase for O2, the O2 concentration of the medium was decreased to 30–40% of the initial value by controlled N2bubbling prior to NADPH addition Below 40–50 lM, the oxygraphic traces curved inward The rates of oxygen uptake were deduced from the slopes of the tangents to the oxygraphic traces, and the contact points of the tangents with the curves were used to determine the O2 concentrations at which O2 uptake proceeds [18] All experiments were repeated two or three times, and the reported results are representative of at least two experiments

Optical spectra Absorption spectra of clear solutions were recorded at room temperature with an Uvikon 930 spectrophotometer In the case of turbid suspensions, a double beam PerkinElmer 557 spectrophotometer was used Reduction was achieved with

a few grains of sodium dithionite The amount of heme b in neutrophil membranes was determined from difference spectra (dithionite-reduced vs oxidized) The molar

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extinc-tion coefficients (De) were 106 mM )1Æcm)1at 425 nm (Soret

Peak) and 21.6 mM )1Æcm)1at 558 nm [21] The amount of

heme b in neutrophil membranes varies from 0.35 to

0.65 nmolÆmg protein)1 depending on preparations In

experiments where reduced heme b was reoxidized by

sequential additions of INT (cf Fig 8), the difference

spectra relative to the Soret band were recorded and the

extent of reoxidation was assessed by the decrease of the

peak of absorbancy by reference to a base-line (see Fig 8)

EPR spectra

EPR spectra were recorded with a X-band Bruker EMX

spectrometer equipped with an Oxford Instruments

ESR-900 continuous flow helium cryostat

R E S U L T S

Effect of NADPH oxidase activation and oxygen

on the rates of INT reductase and O2–production

The experiment illustrated in Fig 1 shows the effects of

arachidonic acid and O2on the rates of electron transfer

from NADPH to cytochrome c and INT Arachidonic acid,

an efficient amphiphile commonly used to elicit the

production of O2 in a cell-free system of NADPH oxidase

activation, was added at increasing concentrations to

neutrophil membranes which were further supplemented

with neutrophil cytosol, GTPcS and ATP After completion

of activation, the rates of cytochrome c reduction and INT reduction were measured either in an aerated medium or in

a N2saturated medium The data were expressed in terms

of lmol e–transferred min)1Æmg membrane protein)1, cor-recting for the fact that cytochrome c is reduced by one electron and INT by two electrons In the aerated medium (Fig 1A), the cytochrome c reductase activity, referred as oxidase activity, and the INT reductase activity both peaked

at a concentration of 1.2–1.3 lmol arachidonic acidÆmg membrane protein)1, the rates of electron transfer being 1.00 lmol and 0.78 lmol e–min)1Æmg membrane pro-tein)1, respectively Both activities differed in their sensitiv-ity to SOD The INT reductase was inhibited  50% by SOD, whereas reduction of cytochrome c was inhibited by

> 95%, indicating that reduction of cytochrome c was essentially due to the superoxide anion O2 generated by the NADPH oxidase

In the oxygen-free medium (Fig 1B) INT was reduced, but not cytochrome c Thus, in contrast to INT, cyto-chrome c does not capture electrons from a redox center of flavocytochrome b The rate of reduction of INT was even higher in anaerobiosis than in aerobiosis (0.94 lmol vs 0.78 lmol e–transferred min)1Æmg membrane protein)1), and nearly the same as the rate of O2 production in aerobiosis This result means that the electron transfer step from NADPH to the redox center from which electrons are captured by INT controls the rate of the overall electron transfer from NADPH to O2

The SOD-sensitive reduction of INT by the activated NADPH oxidase in an aerated medium previously des-cribed and asdes-cribed to the reduction of INT by O2 generated by the oxidase activity of flavocytochrome b [9,11] deserves some comments An alternative explanation

is that INT radicals generated by direct capture of electrons from reduced flavocytochrome b interact with O2 to gen-erate oxidized INT (INTox) and O2 [15,17] according to reaction 1: INT•+ O2« INTox+ O2 Eliminating O2 with SOD displaces the reaction to the right, with formation

of INTox In this mechanism, the superoxide O2 is no longer considered as the product of reduction of O2at the heme level of flavocytochrome b, but rather as the product

of reduction of O2 by INT, and the SOD-dependent inhibition of INT reduction appears to be an indirect effect

On the other hand, the INT radical may generate by dismutation the fully reduced INTredaccording to reaction 2: INT•+ INT•+ H+fi INTred+ INTox

At low concentrations of INT, reaction 1 predominates, whereas at high concentrations of INT reaction 2 (which is second order with respect to the INT concentration) is favored Thus, the balance of INT depends not only on the presence of O2, but also on the concentration of INT This may explain why the SOD-dependent sensitivity of INT reduction fluctuates depending on experimental conditions For example, in a recent report, the extent of inhibition of INT reduction by SOD was limited to 10% [9] compared with 50% in the present paper (Fig 1)

Characterization of the INT radical The reduction of tetrazolium salts into formazan, which involves the overall transfer of two electrons per molecule, proceeds by stepwise addition of individual electrons [22]

Fig 1 Reduction of O 2 and INT by neutrophil membranes activated in a

cell-free system Effect of increasing concentrations of arachidonic

acid Neutrophil membranes (20 lg protein) were incubated at room

temperature with increasing concentrations of arachidonic acid, up to

3 lmolÆmg protein)1, and 5 m M MgSO 4 After 5 min, cytosol (200 lg

protein) was added, together with 0.5 m M ATP and 10 l M GTPcS.

The final volume was adjusted to 50 lL with NaCl/P i and incubation

was continued for a further 5 min The whole sample was transferred to

a photometric cuvette in 2 mL NaCl/P i supplemented with 200 l M

NADPH and either 100 l M cytochrome c (d) or 100 l M INT (j),

depending on the measurement of the oxidase activity (O 2

produc-tion) or the INT reductase activity The assays were carried out in

aerobiosis (A) and in anaerobiosis (B) as described in Experimental

procedures Production of O 2 as a reducing agent was quenched by

addition of 50 lg of superoxide dismutase to the medium of the

photometric cuvette containing either cytochrome c (s) or INT (h)

(A).

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This process involves the formation of a tetrazolinyl free

radical which was found by EPR spectroscopy to be

relatively stable at 25°C in hydrophobic media, even in the

presence of O2 The tetrazolinyl radical can also accumulate

by oxidation of formazan or by disproportionation of a

mixture of formazan and the tetrazolium salt The optical

spectra illustrated in Fig 2A were recorded before and after

addition of a small amount of sodium dithionite to a

solution of INT in a mixture of dimethyl formamide and

water 1 : 1, v/v) The recorded spectrum of oxidized INT

above 400 nm was flat (Fig 2A, trace a) Following

addition of sodium dithionite, an orange color rapidly

developed, with a maximal optical absorbance at 449 nm

(Fig 2A, trace b) In a few minutes, the color changed to red

(Fig 2A, trace c), corresponding to a new spectrum with

two maxima at 500 nm and 550 nm, which was

character-istic of the monoformazan, i.e the fully reduced product of

INT [15,17] In the following aeration of the medium, the

absorbance of the spectral bands decreased, but the shape of

the spectrum remained the same (Fig 2A, trace d), which

means that the fully reduced INT became reoxidized It was

concluded that the transient absorbance at 449 nm

corres-ponded to a partially reduced state of INT, most likely to

the INT radical

The rate of transition from the oxidized state to the fully

reduced state upon addition of sodium dithionite depended

on the medium When the solvent was water, the

red-colored formazan accumulated in a few seconds In contrast, in a mixture of dimethyl formamide and water

of 2 : 1 (v/v), the INT radical was stable for more than

10 min (Fig 2B), the stability of the INT radical increasing with the increase in dimethyl formamide concentration Detergents such as Triton X-100 or SDS at a final concentration of 1% (w/v) also stabilize the INT radical (data not shown) This observation corroborates data showing that the nitroblue tetrazolium (NBT) radical obtained by reduction of oxidized NBT by silver amalgam was stabilized in dimethoxyethane [23] The stabilizing effect

of SDS was not encountered with arachidonic acid which

we routinely used as an activator of the NADPH oxidase The first derivative EPR spectrum at 293 K of INT reduced by sodium dithionite in dimethyl formamide shows a radical structure centered at g¼ 2.00 (Fig 2C, upper trace) The spectrum was characterized by a 10-line pattern, nine of which resemble those of the EPR spectrum

of the 2,3,5 triphenyltetrazolium chloride radical [23] The 10-line pattern of the EPR spectrum of the INT radical could be simulated by assuming a structure containing four equivalent nitrogen atoms and a supernumerary atom with

a spin of 1/2, namely a proton (Fig 2C, bottom trace), with isotropic hyperfine splitting constants of 0.5 mT between nitrogen atoms and 0.75 mT for the supernumer-ary proton

The specific chemical properties of INT and more particularly the stability of its radical in hydrophobic media may explain differences in the efficiency of electron capture by INT depending on experimental conditions of the assay of NADPH oxidase, for example the nature of the detergent used in the cell-free assay or the membrane concentration

Compared effects of the two heme inhibitors, benzylimidazole and bipyridyl on the optical and EPR spectra of hemin and flavocytochromeb

In a preliminary experiment, we found that benzylimidazole and bipyridyl inhibited not only the production of O2 assayed by the SOD-sensitive reduction of cytochrome c, but also the reduction of INT with the same efficiency Half inhibition was obtained with 2–3 mMbipyridyl and 5–7 mM benzylimidazole Inhibition was largely reversed by dilution, indicating that it was not due to denaturation of flavocyt-ochrome b Complementary experiments using optical and EPR spectra were carried out to assess the specificity of the effects of bipyridyl and benzylimidazole on the heme(s) of flavocytochrome b

Hemin was chosen as a model to test by spectral modifications the ability of benzylimidazole and bipyridyl

to react with heme iron Because hemin solutions in detergent are not turbid, absolute spectra of oxidized and reduced hemin in the absence and presence of inhibitors were recorded directly (Fig 3A, trace a, control, and trace

b, presence of benzylimidazole) The difference spectra of dithionite-reduced hemin plus benzylimidazole and dithi-onite-reduced hemin plus bipyridyl minus reduced hemin exemplify typical changes in the spectra consisting of the appearance of well defined peaks at 428 nm, 530 nm and

560 nm in the case of benzylimidazole (Fig 3A, trace c) and

at 437 nm in that of bipyridyl (Fig 3A, trace d) When the same heme inhibitors were added to neutrophil membranes

Fig 2 Evidence for accumulation of a stable INT radical during

reduction of INT by sodium dithionite The INT radical was prepared as

described in Experimental procedures (A) Optical spectra showing the

progressive reduction of INT by sodium dithionite, in a mixture of

dimethyl formamide and H 2 O (1 : 1, v/v), from a fully oxidized state

(a) to a fully reduced state (c) (after 5 min) via a semireduced state

corresponding to the INT radical (b) (after 2 min) The spectrum taken

15 min after aeration (d) has a shape similar to that of the fully reduced

INT, but its size was significantly decreased (B) Spectra of the purified

INT radical in a mixture of dimethyl formamide and H 2 O (2 : 1, v/v),

before (solid line) and after addition of sodium dithionite (dotted line).

(C) Upper trace: first derivative EPR spectrum of the INT radical

recorded at room temperature Microwave power 2 mW; modulation

frequency 100 kHz, modulation amplitude 0.5 mT, microwave

fre-quency 9.660 GHz Bottom trace: simulation of the EPR spectrum

shown in the upper trace was obtained by assuming four equivalent

nitrogen atoms coupled at 0.5 mT and one proton coupled at 0.75 mT.

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in the absence of arachidonic acid, the heme spectrum of

flavocytochrome b was not modified (Fig 3B, trace a)

Neither was it in the presence of arachidonic acid alone in

the absence of inhibitors (Fig 3B, trace b) However, when

the heme inhibitors were added to the neutrophil

mem-branes in the presence of arachidonic acid used at a

concentration that elicited maximal oxidase activity,

signi-ficant spectral modifications were recorded These

modifi-cations consisted in a decrease of the Soret peak

accompanied by a slight blue shift and in a decrease of the

a peak (Fig 3B, trace c, benzylimidazole, and trace d,

bipyridyl) Moreover, addition of benzylimidazole and

bipyridyl resulted in opposite modifications of the sizes of

the a and c peaks of heme b Benzylimidazole decreased the

a/c peak ratio from 4.5 to 3.4 whereas bipyridyl increased it

from 4.5 to 6.1, suggesting different types of constraint

applied to the hemes by the two inhibitors

Binding of benzylimidazole to the heme iron of hemin

and to the heme iron of purified flavocytochrome b was

assessed by EPR spectroscopy (Fig 4) Addition of

benzy-limidazole to hemin resulted in the decrease of the high spin

signal at g¼ 6.0 (Fig 4, trace d), characteristic of the

pentacoordinated form of the iron atom of the heme, and in

the concomitant emergence of a low spin signal with

components at g1¼ 2.97 and g2¼ 2.25 (trace e) Purified

flavocytochrome b in detergent displayed a mixture of

penta- and hexacoordinated forms of heme b (Fig 4, trace

a) The high spin signal at g¼ 6.0 similar to that of hemin accounted for the pentacoordinated form of the heme iron The hexacoordinated form was represented by two low spin

g1signals at g¼ 3.28 and g ¼ 2.85 The g2components are probably associated at g¼ 2.20 The signal at g ¼ 4.3 was due to adventitious ferric species [24] Thus, even in the absence of arachidonic acid, a fraction of purified flavocyt-ochrome b is pentacoordinated and capable of reacting with

O2 or with heme ligands The high spin fraction was significantly increased by addition of 100 lMarachidonic acid, whereas the low spin signals were totally erased (data not shown) in accordance with previous results [18] The high spin signal of purified flavocytochrome b (see control trace a) was decreased by addition of 25 mM benzylimidaz-ole (trace b), and nearly abolished at 50 mM benzylimidaz-ole (trace c), a concentration which also fully inhibited the NADPH oxidase activity Concomitantly with the disap-pearance of the high spin signal at g¼ 6.0, a low spin signal with components g1 and g2 at g¼ 2.97 and g ¼ 2.25 emerged at positions similar to those observed in the case of the hemin/benzylimidazole complex (trace e), probably due

to the binding of benzylimidazole as an axial ligand to the heme iron in hemin or in flavocytochrome b The two low spin signals g1at g¼ 3.28 and g ¼ 2.85 initially present in purified flavocytochrome b were not altered upon addition

of 50 mMbenzylimidazole This behavior is reminiscent of the absence of effect of benzylimidazole on the optical spectrum of resting neutrophil membranes in the absence of arachidonic acid Thus, the fraction of purified flavocyto-chrome b characterized by low spin signals contains a hexacoordinated heme iron unable to react with benzylim-idazole; this fraction, calculated by integration, represents roughly half of the total amount of flavocytochrome b

Fig 4 Effect of benzylimidazole on the EPR spectra of isolated cytochrome b and hemin Traces a–c are EPR spectra of purified flavo-cytochrome b (45 l M ) in solution in 20 m M P i , 20% glycerol, 0.5 M

NaCl and 0.1% Triton X-100, pH 7.4 Trace a corresponds to control flavocytochrome b, trace b to flavocytochrome b treated with 25 m M

benzylimidazole and trace c to flavocytochrome b treated with 50 m M

benzylimidazole Traces d and e correspond to hemin (1 m M ) in DMF untreated and treated with 50 m M benzylimidazole, respectively (A) and (B) show the high spin and low spin regions of the EPR spectra, respectively.

Fig 3 Effects of benzylimidazole and bipyridyl on optical spectra of

hemin and membrane bound flavocytochrome b (A) Traces a1 and a2:

absolute spectra of oxidized and dithionite-reduced hemin (10 l M ),

respectively Traces b1 and b2: absolute spectra of oxidized and

dithionite-reduced hemin in the presence of 5 m M imidazole Trace c:

difference spectrum of reduced hemin plus 5 m M benzylimidazole

against reduced hemin (10 l M ) Trace d: Difference spectrum of

reduced hemin plus 10 m M bipyridyl against reduced hemin (20 l M ).

(B) Traces a–d: difference spectra (dithionite-reduced minus oxidized)

at room temperature of neutrophil membranes in NaCl/P i

(1 mg proteinÆmL)1 equivalent to 0.65 nmol heme b) Trace a,

control; trace b, membranes supplemented by arachidonic acid

(1.3 lmolÆmg protein)1); trace c, reduced membranes plus arachidonic

acid treated for 5 min with 40 m M bipyridyl; trace d, reduced

membranes plus arachidonic acid treated for 5 min with 25 m M

benzylimidazole.

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Dependence of NADPH oxidase inhibition by 5-deaza FAD

on the activation state of flavocytochromeb

The effect of 5-deaza FAD, a flavin analog and an obligate

two-electron donor [25–27] was tested on the NADPH

oxidase activity and the INT reductase activity of

flavocyto-chrome b in the cell-free system (Fig 5) Its inhibitory effect

depended on the step at which it was added to the medium

When 5-deaza FAD was added to the activated cell-free

system, the NADPH oxidase and the INT reductase were

hardly inhibited On the other hand, when 5-deaza FAD

was preincubated with neutrophil membranes together with

arachidonic acid 5 min prior to addition of the other

components of the activation system, namely cytosol,

GTPcS and ATP, both the elicited NADPH oxidase and

INT reductase activities were inhibited efficiently

(Ki¼ 25 lM), and the extent of inhibition was the same

for the two activities The inhibition caused by 5-deaza

FAD was prevented by addition of a 10· excess of FAD

(data not shown) Together these results suggest that

arachidonic acid induces by itself some structural

modifica-tions in flavocytochrome b which result in the release of the bound FAD and its replacement by 5-deaza FAD When the oxidase is fully activated, these modifications do not occur any more It is noteworthy that, in contrast to the flavin analogs, the heme inhibitors benzylimidazole and bipyridyl were equally effective when added either to the activated cell-free system or to the membranes before NADPH oxidase activation

Effect of 5-deaza FAD and heme inhibitors

on the reduced state of flavocytochromeb

in activated neutrophil membranes

In the experiment of Fig 6 conducted in anaerobiosis, 5-deaza FAD was preincubated with neutrophil membranes and arachidonic acid 5 min before the addition of cytosol, GTPcS and ATP, a condition required for the optimal inhibitory effect of the flavin analog on the oxidase activity

In the control assay (Fig 6, trace a), addition of NADPH resulted in an abrupt rise of heme b reduction, followed by a plateau which corresponded to 40–50% of the full reduction obtained with sodium dithionite After the reduction plateau had been reached, 10 nmol O2(dissolved in buffer) were added, which resulted in an abrupt, but limited, reoxidation of heme b, rapidly counteracted by the electron flux issued from NADPH After a new redox equilibrium had been attained, two other redox cycles were initiated by addition of 5 and 10 nmol of INT Reoxidation of reduced heme b with 10 nmol INT was twice that with 5 nmol INT, indicating proportionality over this range of INT concen-tration Moreover, 5 nmol INT (a mediator which is reduced by a pair of electrons) are able to oxidize the same amount of reduced heme b as 10 nmol O2, which speaks in favor of the idea that reduced heme b is the source of electrons for both INT and O2 If the major source of electrons for INT were reduced FAD, assuming a back flow

of electrons from reduced heme b to FAD, the above stoichiometry would be different The experiment was completed by addition of sodium dithionite in limiting amounts just sufficient to reduce  95% of the heme components of flavocytochrome b Addition of 10 nmol O2 (dissolved in buffer) followed by 10 nmol INT resulted in oxidation cycles of heme b similar to those obtained in the presence of NADPH alone as reducing agent

Trace b (Fig 6) refers to the effect of 5-deaza FAD used

at a concentration that inhibited 80% of the oxidase activity The rate and the extent of the NADPH-dependent reduction of heme b were both largely decreased This explains why the redox cycles initiated by addition of

O2and INT were smaller in size, compared to the control (trace a); yet the dithionite-reduced heme b was reoxidized

by O2and INT to virtually the same rate and extent as in the control, despite the block imposed by 5-deaza FAD at the level of the flavin redox center of flavocytochrome b With a higher concentration of 5-deaza FAD, which inhibited

> 95% of the NADPH-dependent reduction of heme b, INT and O2were still able to reoxidize the dithionite reduced heme b (trace c, Fig 6) The two later traces (d and e, Fig 6) refer to the action of the heme inhibitors, benzylimidazole and bipyridyl Both inhibitors strongly interfered with the rate and extent of reoxidation of the heme b reduced in the presence of NADPH Following addition of sodium dithi-onite, the cycles of reoxidation of heme b initiated by O and

Fig 5 Dose-dependent inhibition of the NADPH oxidase and INT

reductase activities of neutrophil membranes by the flavin analog 5-deaza

FAD Oxidase activation was performed as described in Experimental

procedures and in the legend of Fig 1 5-deaza FAD was added with

arachidonic acid and MgSO 4 to the membrane suspension (20 lg

protein) After 5 min at room temperature, oxidase activation was

elicited by addition of cytosol and GTPcS After an additional 5 min

incubation, the oxidase activity was measured as O 2 production (d)

and the INT reductase activity by the rate of reduction of INT (j).

A parallel experiment was carried out in which 5-deaza FAD was

incubated for 5 min with the membrane suspension after the cell-free

activation of the NADPH oxidase; O 2 production (s), INT reductase

activity (h).

Trang 7

INT were also significantly diminished, compared to the

control (trace a) In summary, using the flavin analog

5-deaza FAD and the heme inhibitors, bipyridyl and

benzylimidazole, to limit or to block the flux of electrons

along the redox centers of flavocytochrome b, it is possible

to demonstrate that INT is able to capture electrons from

the heme components of flavocytochrome b

Kinetics of inhibition of the oxidase activity by INT and bipyridyl

As INT appeared to capture electrons from the hemes of flavocytochrome b, we asked whether INT could compete with O2 Activated neutrophil membranes in a cell-free system in the presence of cytosol, GTPcS and arachidonic acid were placed in an oxygraphic cuvette containing the NaCl/Pimedium whose O2concentration had been previ-ously lowered to  80 lM by controlled bubbling of N2 Then, O2uptake was initiated by addition of a saturating concentration of NADPH (250 lM) Below 80 lMO2, the oxygraphic traces curved inwards In the portion of the traces corresponding to O2concentrations ranging between

80 lMand 20 lM, the rates of O2uptake were deduced from the slopes of tangents at different concentrations of O2 The assay was repeated with INT added to the medium at increasing concentrations In the absence of INT, the reciprocal plots corresponded to a straight line from which a

Kmvalue of 25 lMfor O2could be deduced (Fig 7A) At increasing concentrations of INT, the plots corresponded also to straight lines that intersected the 1/v axis at a common intercept, which was the same as that of the control curve, and the apparent Kmfor oxygen increased in proportion to the increase in INT concentration These features are typical of a competitive inhibition The Kifound for INT was 30 lM, a value which is of the same order as the Kmfound for O2[13]

Assuming that capture of electrons from reduced heme b

by INT was responsible for the competitive effect of INT on the oxidase activity of flavocytochrome b, it was inferred that a heme b inhibitor should competitively inhibit in anaerobiosis the reduction of INT by membranes of neutrophils activated in a cell-free system This was in fact the case with bipyridyl as shown in Fig 7B A similar competitive inhibition of O2uptake by bipyridyl was found when activated neutrophil membranes were incubated with NADPH in an aerated medium (Fig 7C) In both cases (Fig 7B,C) the calculated Kifor bipyridyl were the same, namely 2 mM± 0.2 mM Twice higher values for Kiwere obtained with benzylimidazole (data not shown)

INT-dependent reoxidation of dithionite-reduced heme b

in flavocytochromeb and dithionite-reduced hemin

In this experiment, we followed the stepwise oxidation of reduced flavocytochrome b and reduced hemin by sequen-tial additions of small amounts of INT Neutrophil membranes pretreated by arachidonic acid were placed in photometric cuvettes under a flow of nitrogen It is known that pretreatment of membranes by arachidonic acid modifies the spin state of heme b [18], but is not sufficient per se to elicit the oxidase activity of flabocytochrome b

A parallel spectrophotometric assay was carried out with hemin Hemin and flavocytochrome b were reduced to

 95% by the addition of a limited amount of sodium dithionite To reoxidize hemin and the heme component of flavocytochrome b, INT was added by small increments to the anaerobic cuvettes Absorbance was recorded using a double wavelength spectrophotometer, between 380 nm and 480 nm for hemin and between 400 nm and 465 nm for flavocytochrome b, corresponding to the Soret peak of the two pigments Reoxidation of hemin or the heme of

Fig 6 Effect of 5-deaza FAD, bipyridyl and benzylimidazole on the O 2

and INT-dependent reoxidation of reduced heme b in activated

neutro-phil membranes Activated membranes (2 mg protein in 1.7 mL,

equivalent to 0.70 nmol heme b) in the cell-free system (arachidonic

acid, cytosol and GTPcS) were placed in a photometric cuvette The

medium (1.7 mL) was made anaerobic as described in Experimental

procedures The following compounds were injected into the medium

in minimal volumes in the following sequence: NADPH, 1 lmol; O 2 ,

10 nmol (dissolved in buffer); INT, 5 and 10 nmol; sodium dithionite,

1 lmol Trace a, control membranes; trace b, membranes

preincu-bated for 10 min with 40 l M 5-deaza FAD and arachidonic acid

fol-lowed by addition of cytosol and GTPcS; trace c, same conditions as in

trace b except that 5-deaza FAD was used at 80 l M ; trace d, same

conditions as in trace a with 10 m M benzylimidazole (BI) added to the

cuvette after reduction of heme b by NADPH; trace e, same conditions

as in trace a with 10 m M bipyridyl (Bipy) added to the cuvette after

reduction of heme b by NADPH After full reduction of heme b with

sodium dithionite, further cycles of oxidation were initiated by

addi-tion of O 2 and INT (respectively 10 and 5 nmol).

Trang 8

flavocytochrome b was assessed by the decrease of the

absorbance (Fig 8, insert) Up to 80% heme reoxidation, a

linear relationship between the amount of added INT and

the absorbance decrease was observed (Fig 8) Above 80%

heme reoxidation, the curves departed from linearity The

two dose–response curves for hemin and flavocytochrome b

were virtually superimposable By extrapolation of the

linear portions of the curves to the abscissa, it could be

calculated that 0.45–0.48 mol INT was needed to reoxidize

1 mol hemin or 1 mol heme of flavocytochrome b, which is

consistent with the stoichiometry of 2 e–captured per INT

molecule in both cases Thus, like reduced hemin, the

reduced heme of flavocytochrome b is able to donate

electrons to INT A back transfer of electrons from reduced

heme b to FAD should not be significant because, as noted

above, the oxidase activity of neutrophil membranes treated

with arachidonic acid is dormant in the absence of cytosolic

factors [18] and, consequently, electron transfer between the

redox centers of flavocytochrome b is maintained at a very

low level In summary, the value of the stoichiometric ratio

of reduced INT to oxidized heme suggests that under our

experimental conditions electrons are essentially transferred

from the heme components of flavocytochrome b to INT

D I S C U S S I O N

The postulate that the flavocytochrome b component of the

neutrophil NADPH oxidase has the potential to display a

diaphorase activity stems from two different observations

First, artificial disruption of the electron transfer chain of

flavocytochrome b by detergents allows capture of electrons

upstream of heme b [8] Second, a mutated

flavocyto-chrome b from a CGD (91X+) patient, that was unable to

reduce O2into O2, was still able to carry electrons from

NADPH to INT This latter observation led to the belief

that INT is a suitable electron acceptor from the reduced

FAD component of flavocytochrome b and thereby a suitable probe of the diaphorase activity of flavocyto-chrome b [9] There were, however, in the meantime, reports that pointed to the rather complex behavior of INT [15,17]

We therefore decided to determine by classical techniques, using specific inhibitors of the electron flux in flavocyto-chrome b, which redox components interacted with INT The flavin analog, 5-deaza FAD was used to inhibit the flux

of electrons at the FAD level On the other hand, two efficient heme inhibitors, bipyridyl and benzylimidazole, were selected after validation by optical and EPR spectro-metric tests

The experiments described here led us to conclude that INT is able to capture electrons from the heme b components of the neutrophil flavocytochrome: (a) in activated membranes maintained in anaerobiosis, heme b reduced by NADPH was reoxidized by INT as efficiently

as by O2 (Fig 6) Reoxidation of heme b was insensitive

to 5-deaza FAD, which contrasted with the inhibitory effect of this compound on the reduction of heme b by NADPH (Fig 6) This result, which agrees with the well recognized function of 5-deaza FAD as a flavin inactive analog, rules out the possibility of a back reaction from heme to flavin and then from flavin to INT; (b) INT was able to reoxidize hemin reduced by sodium dithionite (Fig 7) As FAD was absent in this experiment, electrons were directly transferred from hemin to INT The comparative titrations by INT of dithionite-reduced hemin and dithionite-reduced heme b of flavocytochrome b ended with the same stoichiometry of roughly 0.5 mol INT reduced by 1 mol hemin or by 1 mol heme b in flavocytochrome b; (c) INT was able to compete with O2

in an aerobic medium, and also with the heme inhibitor, bipyridyl, in anaerobiosis (Fig 7) These results led us to conclude that the heme components of flavocytochrome b interact directly with INT

Fig 7 Kinetics of inhibition of the elicited NADPH oxidase activity in a cell-free system (A) and (C), aliquots of neutrophil membranes (150 lg protein in A) activated in the cell-free system were placed in the oxygraphic cuvette containing 1.5 mL NaCl/P i supplemented with different fixed concentrations of INT in A (d, zero; s, 30 l M ; j, 60 l M ; h, 90 l M ; _, 120 l M ) and bipyridyl in C (j, zero; d, 3 m M ; h, 7 m M ; s, 15 m M ) The initial concentration of O 2 (230 l M ) was decreased by about two-thirds by controlled bubbling of nitrogen prior to the assay of O 2 uptake initiated

by addition of 250 l M NADPH The rate of O 2 uptake was calculated from the slopes of the tangents to the oxygraphic traces (B) Activated membranes (40 lg protein per assay) were placed in a closed photometric cuvette containing 1 mL of a medium previously made anaerobic by N 2

bubbling The medium consisted of NaCl/P i supplemented with 250 l M NADPH and different concentrations of INT ranging from 20 to 200 l M The rate of INT reduction recorded at 500 nm was calculated using a molar extinction coefficient of 11 m M )1 Æcm)1 Bipyridyl was used at the following concentrations: j, zero; d, 3 m M ; h, 7 m M ; s, 15 m M

Trang 9

Theoritically, INT might accept electrons from reduced

FAD in activated flavocytochrome b inasmuch as the FAD

binding site is believed to be located in the relatively

hydrophilic C-terminal half of the gp91phox subunit of the

flavocytochrome Experimental data show that this is not

the case, which raises the question of the capacity of INT to

probe specifically the diaphorase activity of neutrophil

NADPH oxidase It is however, not excluded that under

specific conditions, for example the presence of a detergent

or of a mutation such as Arg54Ser [14], the structural

arrangement of the FAD binding domain of

flavocyto-chrome b is modified, resulting in a loss of interaction of

FAD with heme 1 and in a facilitated access of INT to FAD Along this line, one may recall that a peculiar behavior of FAD in flavocytochrome b was recognized in the past [28], and explained in terms of a kinetic barrier between flavin and heme b A peculiar structural arrange-ment of the peptide chain in the FAD region of flavocyto-chrome b, possibly related to the oligomerization of the protein, might be responsible for its very unusual properties Finally, we would like to point out that some of the unusual properties of INT might be due to the stabilization of its radical in hydrophobic media, for example the lipid phase of membranes

A C K N O W L E D G E M E N T S

We thank V Massey for the gift of 5-deaza FAD, J Willison for careful reading of the manuscript and J Bournet-Cauci for excellent secretarial assistance This work was supported by funds from the Centre National

de la Recherche Scientifique, the Commissariat a` l’Energie Atomique, the Universite´ Joseph Fourier–Grenoble I, and the Association pour la Recherche sur le Cancer (9996).

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Fig 8 Effects of INT on reoxidation of dithionite-reduced hemin and

dithionite-reduced flavocytochrome b Neutrophil membranes in

NaCl/P i (2 mg proteinÆmL)1; 0.60 nmol heme bÆmg protein)1, total

volume 1.7 mL) were preincubated for 10 min at room temperature

with arachidonic acid, 1.5 lmolÆmg protein)1 (j) A solution of

freshly prepared hemin was used at the concentration of 10 l M in

NaCl/P i supplemented with 1% Triton X-100 (s) Neutrophil

membranes or hemin were placed in a photometric cuvette The

medium had previously been made anaerobic by bubbling N 2 , and

maintained anaerobic during the optical assays as described in

Experimental procedures After a few minutes of anaerobiosis, small

amounts of sodium dithionite solution were added to attain a level of

reduction of heme b and hemin of 90–95% Sequential additions of

small aliquots (0.2 lL) of a solution of INT previously made anaerobic

by N 2 bubbling were injected to the medium until heme b and hemin

were fully reoxidized After each addition the difference spectra

(reduced minus oxidized) were recorded As shown in the insert in the

case of flavocytochrome b, the loss of absorbance was measured

between the peak of absorbance of the fully reduced spectrum and a

base-line drawn between the isosbestic point at 416 nm and 450 nm.

The results were normalized in terms of percent of reduced heme and

plotted against the calculated ratio of added INT to oxidized heme

(mol/mol) A similar procedure was used in the case of hemin.

Trang 10

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26 Spencer, R., Fisher, J & Walsh, C (1976) Preparation, char-acterization, and chemical properties of the flavin coenzyme analogues 5-deazariboflavin, 5-deaza-riboflavin 5¢-phosphate, and 5-deazariboflavin 5¢-diphosphate, 5¢-5¢ adenosine ester Biochem-istry 15, 1043–1053.

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