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Perhaps the best-studied family of interfacial enzymes Keywords bilayer; catabolism; cholesterol oxidase; GMC oxidoreductase; interfacial enzyme; lipid phase; macrolide biosynthesis; mem

Trang 1

Cholesterol oxidase: physiological functions

Joseph Kreit1and Nicole S Sampson2

1 Laboratory of Biochemistry and Immunology, Department of Biology, Faculty of Sciences, Mohammed V University, Rabat, Morocco

2 Department of Chemistry, Stony Brook University, NY, USA

Introduction

3b-Hydroxysteroid:oxygen oxidoreductase (EC 1.1.3.6),

commonly known as cholesterol oxidase (ChOx), is

a flavoenzyme that catalyzes the oxidation and

isomeri-zation of cholesterol to cholest-4-en-3-one and has

been well characterized structurally and chemically (see

the first review of this miniseries) The enzyme is

extra-cellular and occurs in a secreted form and⁄ or a

cell-surface-associated form, depending on the producer

microorganism and growth conditions Both forms are

products of the same gene The secreted form is a

solu-ble, globular protein, and the X-ray crystal structures

[1–3] revealed that it is essentially composed of two

fused domains: the flavin-binding domain and the

substrate-binding domain An important aspect of the

catalysis carried out by this enzyme is the nature of its

association with the lipid bilayer that contains the sterol

substrate Efficient catalytic turnover is affected by the

association of the protein with the membrane as well as

the solubility of the substrate in the lipid bilayer In this review, we discuss the binding of ChOx to the lipid bilayer, its turnover of substrates presented in different physical environments and how these conditions affect substrate specificity Defining substrate specificity with respect to these parameters is important for understand-ing the physiological functions of the enzyme in bacte-rial metabolism and perhaps in pathogenesis We will begin with an overview of interfacial membrane kinetics and how they pertain (a) to the reaction catalyzed by ChOx and (b) to understanding the implications for investigating substrate specificity

Interfacial kinetics

An interfacial enzyme is a protein that binds tran-siently to the membrane surface during catalysis Perhaps the best-studied family of interfacial enzymes

Keywords

bilayer; catabolism; cholesterol oxidase;

GMC oxidoreductase; interfacial enzyme;

lipid phase; macrolide biosynthesis;

membrane; sterol; virulence

Correspondence

N S Sampson, Department of Chemistry,

Stony Brook University, New York,

11794-3400, USA

Fax: +1 631-632-5731

Tel: +1 631-632-7952

E-mail: nicole.sampson@stonybrook.edu

(Received 26 July 2009, revised 2 September

2009, accepted 10 September 2009)

doi:10.1111/j.1742-4658.2009.07378.x

An important aspect of catalysis performed by cholesterol oxidase (3b-hy-droxysteroid oxidase) concerns the nature of its association with the lipid bilayer that contains the sterol substrate Efficient catalytic turnover is affected by the association of the protein with the membrane as well as the solubility of the substrate in the lipid bilayer In this review, the binding of cholesterol oxidase to the lipid bilayer, its turnover of substrates presented

in different physical environments, and how these conditions affect sub-strate specificity, are discussed The physiological functions of the enzyme

in bacterial metabolism, pathogenesis and macrolide biosynthesis are reviewed in this context

Abbreviations

ChOx, cholesterol oxidase; GMC oxidoreductase, glucose-methanol-choline oxidoreductase; CAMP reaction, Christle–Atkins–Munch– Petersen reaction.

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is the phospholipase A2 family and extensive

develop-ment of the kinetic model derives from its study [4]

The two key components of the interfacial kinetic

model are (a) that the enzyme freely dissociates from

the membrane surface and (b) that the important

measure of substrate quantity is its mole fraction in

the membrane, not bulk solution concentration

(Scheme 1) Analagous to steady-state measurements

with soluble substrates, the initial velocity dependence

on the mole fraction of substrate may be determined

as long as the fraction of enzyme bound to the surface

is known Typically, the initial rate dependence on the

mole fraction is hyperbolic and is fit to the Michaelis–

Menten equation Km* is the mole fraction of substrate

at which the initial velocity is half-maximal, and kcat*

is the first-order rate constant for turnover when all

enzyme molecules are bound to the membrane and

sat-urated with substrate The paradigm for understanding

phospholipase interfacial kinetics is applicable to

ChO-xs Although cholesterol desorbs from membranes

much more rapidly than phospholipids, the rate of

cholesterol desorption is about five orders of

magni-tude slower than the catalytic turnover rate of ChOx

measured in a variety of membranes Therefore, the

enzyme does not capture free cholesterol from aqueous

solution Rather, ChOx associates with lipid bilayers,

and binds cholesterol from the membrane

Cholesterol mixed into membranes does not behave

as an ideal solute In the ideal case, as more

choles-terol is added to a single phospholipid component

membrane (binary mixture), the chemical activity of

the cholesterol increases in proportion to the mole

fraction However, the activity can change nonlinearly,

depending on the structure of the lipids mixed with

cholesterol and the liquid phase present In other

words, the chemical activity of the cholesterol

sub-strate is not only dependent on its mole fraction; it

also depends on the probability that cholesterol will

leave the membrane [5]

The rate at which cholesterol is desorbed from the membrane is affected as intermolecular packing changes within the membrane The liquid membrane is essen-tially a solvent for the substrate, and changing the nature of the solvent (for example, the structures of the lipid acyl chains) results in a change in the desorption rate The molecular interactions between lipids and cholesterol determine the free energy of the cholesterol

in the lipid Consequently, the equilibrium between substrate binding to the enzyme and substrate residing

in the membrane changes as the structure of the lipid membrane changes Lipids with saturated acyl chains have a greater affinity for cholesterol than lipids with unsaturated acyl chains Head groups affect the affinity

of the membrane for cholesterol in the order sphingo-myelin > phosphatidylserine > phosphatidylcholine > phosphatidylethanolamine [6] Thus, the enzyme’s apparent specificity for different sterols can depend on the molecular interactions between lipids and substrates

as much as it depends on interactions between substrate and enzymes This relationship was recognized early and forms the basis of using ChOx to probe the physiological partitioning of cholesterol in biological membranes [7–13] Moreover, ChOx has insecticidal properties against Coeloptera larvae, agricultural pests (covered in the third review of this miniseries), and has been undergoing development for use in agricultural crop treatments [14] The efficacy of the treatment depends on the relative specificity of the enzyme for pest membranes versus plant membranes [15]

Membrane effects on cholesterol oxidase activity

Generally, lipid bilayers exist in a gel phase or a liquid phase in the absence of cholesterol Lipids above their melting temperature have greater lateral mobility within the lipid bilayer and hence behave more like liquids than solids In membranes composed of lipids with satu-rated acyl chains (e.g dipalmitoylphosphatidylcholine), introduction of cholesterol into the liquid phase results

in an increase in membrane order to form a phase that is still liquid, that is, the lipids still have lateral mobility, but there is a higher degree of order The cholesterol constrains the saturated lipid acyl chains to an S-trans conformation that limits disorder in the center of the lipid bilayer This liquid-ordered state separates from the cholesterol-free liquid-disordered state, and the two phases can co-exist [16–18] In addition, as a result of multibody molecular interactions that occur at critical cholesterol mole fractions, ChOx activity may be decreased or increased nonproportionately with the mole fraction within a single-phase region [6,19–21]

E + vesicle Kd E*+ S K E*• S E*+ P

m kcat

Vesicle surface

Scheme 1 Model for interfacial Michaelis–Menten kinetics at a

membrane surface E, free enzyme; E*, membrane-bound enzyme;

kcat*, interfacial first-order rate constant in min)1; Km*, interfacial

Michaelis constant in units of mole fraction; P, product in the

mem-brane; S, substrate in the membrane.

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Thus, increasing the mole fraction of cholesterol can

perturb the physical state of the membrane and alter the

affinity of the membrane for cholesterol Therefore, the

enzyme’s apparent substrate specificity will depend on

the lipid composition and substrate mole fraction at

which the enzyme activity is determined

Further complicating the determination of ChOx

substrate specificity, many studies are performed in

detergent micelles [22,23] Early studies demonstrated

that the rate of cholesterol oxidation was highest with

nonionic detergent micelles [e.g Triton X-100

(polyeth-ylene glycol octylphenyl ether) or Thesit (polyeth(polyeth-ylene

glycol monododecyl ether)] containing cholesterol

[24,25] The elements of the kinetic model are the same

with detergent micelles The enzyme must associate

with the surface, and the mole fraction of cholesterol

is the important element The free energy of

interac-tion of cholesterol with the detergent can have a large

effect on the apparent catalytic activity of the enzyme

For example, no turnover is detected with

cetyltri-methylammonium bromide⁄ cholesterol micelles, whereas

at the same concentration, pH and temperature, Triton

X-100⁄ cholesterol micelles are oxidized readily [24] In

addition, the Km values widely reported using Triton

X-100⁄ cholesterol micelles are actually apparent Km

values that include a term for binding to the micellar

surface The consequence of this is that many changes

to active-site binding have no apparent effect on the

Km [26–28] because this kinetic term is dominated by

the micelle binding

The corollary is that comparing different substrates in

detergent micelles is only useful for understanding

substrate specificity in the presence of detergent The

free energy of association between substrates and

detergent can dictate the apparent preference of the

enzyme for different steroids For example, ChOx is

two- to four-fold more specific for cholesterol over

sitosterol and stigmasterol in detergent micelles, but

shows equal specificity for all three sterols in

dioleoylphosphatidylcholine⁄ sterol vesicles [29] For

biotechnology applications, such as measuring

concen-trations of sterols (discussed in the third review of this

miniseries), the use of detergent micelles makes sense

However, for understanding the physiological function

or the potential role of ChOx in pathogenesis, the

specificity must be studied in the context of lipid

membranes

Cholesterol oxidase–membrane

interactions

The association mechanism of interfacial catalysis

(Scheme 1) requires that the substrate-binding site is

oriented towards the lipid bilayer and is in contact with it X-ray crystal structures [2,3,30] of the soluble enzyme in the absence of lipid identified the face that must be oriented towards the membrane containing the substrate The Streptomyces and Rhodococcus equi (misclassified as Brevibacterium sterolicum) enzymes have been the focus of these studies because they are amenable to expression, mutagenesis and high-resolu-tion crystallography Their mechanism and structures are nearly identical [31] On this face, the substrate-binding cavity is protected from solvent by a protein loop comprising residues 79–83

The role of this surface loop was investigated by deletion of the five amino acid residues, Ser79, Phe80, Leu81, Trp82 and Leu83 at the tip of the loop [32] (Fig 1) The Kd for binding to phosphati-dylcholine-cholesterol vesicles was not affected in the mutant However, the mutant k*cat⁄ Km* value was reduced nearly 3000-fold with phosphatidylcholine-cholesterol vesicles (present in a 1 : 1 ratio) as sub-strate These experiments were interpreted to suggest that the loop is required for cholesterol to bind to the enzyme, but not for binding to the membrane The loop is amphipathic, and the four tip residues deleted are hydrophobic groups that must pack with the eight carbons of the cholesterol side chain in the open form of the loop (Fig 1) In the Streptomyces enzyme, the region comprising residues 78–87 adopts

a small amphipathic helical turn with hydrophobic residues directed towards the active site cavity and hydrophilic residues directed towards the external surface of the molecule [33] In this conformation, the active site is covered and thus aggregation of the protein at its active site is prevented Upon substrate binding, hydrophobic interactions between the hydrophobic residues and cholesterol minimize energy loss

From inspection of the X-ray crystal structures, con-formational changes must accompany substrate bind-ing The position of the active-site loop upon binding

to model membranes (lipid vesicles) was determined using fluorescence quenching [34] In this study, a cys-teine was introduced into the loop at position 81 of the ChOx from R equi (B sterolicum) and labeled with acrylodan, an environmentally sensitive fluorescence probe Modeling the acrylodan-labeled cysteine as an extended chain of the loop revealed that the backbone

of this loop does not penetrate into the lipid bilayer but interacts with the head groups of the lipid bilayer This experiment suggests that the enzyme sits on the membrane surface Slotte has also demonstrated that tetramethylrhodamine-labeled ChOx associates with cholesterol⁄ dimyristoylphosphatidylcholine monolayers

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[35] This surface binding is consistent with vesicle lysis

studies which demonstrated that binding of the enzyme

to membrane surfaces does not disrupt the membrane

[36] The association of ChOx with lipid bilayers,

together with the partitioning of cholesterol into its

active site, does not alter the bilayer sufficiently to

allow pore formation and consequent leakage of dye

encapsulated in vesicles However, conversion of

cho-lesterol to cholest-4-en-3-one does increase membrane

permeability by expansion (actually decondensation) of

the lipid bilayer

Using the fluorescently labeled enzyme, binding to a

variety of lipid vesicle types was monitored Enzyme

binding to the membrane is insensitive to charge and

appears to be driven by hydrophobic interactions [34]

Compared with phospholipase A2 binding to anionic

membranes, the binding affinity of ChOx is very weak,

which means that many studies have been performed

without saturating the membrane surface with enzyme

and differences between substrate preparations may

simply reflect differences in binding affinity

Enzyme substrate specificity

After enzyme binding to the membrane surface, sterol must bind in the enzyme active site for catalysis to occur There are few studies of different sterols in lipid bilayer environments An absolute requirement for activity is the presence of the 3b-hydroxy group

on the steroid framework Modification of the cho-lesterol side chain, ranging from no side chain (e.g 5-androstene-3b-ol) to a branched side chain (e.g sitosterol), has little effect on substrate specific-ity when the steroids are monolayers [37] or in dioleoylphosphatidylcholine⁄ sterol unilamellar vesicles [29] This lack of specificity is in distinct contrast to substrate specificity studies in detergent micelles or with propan-2-ol co-solvent that demonstrates a specificity for cholesterol over sitosterol, androsten-3b-ol and related steroid structures (see the third review of this miniseries) [38,39]

Two types of model membrane have been used to study ChOx specificity for cholesterol in different

mem-A

Fig 1 3D structure of ChOx showing the

active-site loop (A) Ribbon cartoon of

Strep-tomyces ChOx (1MXT [3]) with

epiandros-terone modeled into the active site (shown

in cyan) The FAD cofactor is shown in

yel-low The active-site loop that must move to

allow substrate binding is shown in blue (B)

Stick atomic representation of the

Strepto-myces ChOx active-site loop from (A) in the

same orientation (C) Stick atomic

represen-tation of active-site loop from

Rhodococ-cus equi (formerly Brevibacterium

sterolicum) ChOx (1COY) [2] The entire

Rhodococcous ChOx structure was overlaid

with the Streptomyces ChOx structure and

the loops in (B) and (C) are depicted in the

same enzyme orientations Side chains for

which there is no electron density were

modeled as alanines The residues that

were deleted in Sampson et al [32] are

shown with a magenta carbon backbone.

This figure was contructed using PYMOL

[103] [Correction added on 22 October

2009 after first online publication: in

Fig 1B,C the colours and labelling have

been amended.]

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branes, namely unilamellar vesicles and monolayers.

The specificities observed follow the same trend In both

cases, the activities of the enzyme correlate with the

chemical activity of cholesterol in the lipid membrane

The more favorable the packing interactions between

cholesterol and lipid, the lower the catalytic activity of

the enzyme For example, the kcat*⁄ Km*with cholesterol

mixed with dioleoylphosphatidylcholine is twofold

higher than the kcat*⁄ Km* with cholesterol mixed with

dipalmitoylphosphatidylcholine in similar mole fraction

regimes [40] When the dipalmitoylphosphatidylcholine

is substituted with sphingomyelin, the kcat*⁄ Km* value

shows a 40-fold decrease [41,42] In addition, it is not

possible to saturate the enzyme with cholesterol That is,

the initial velocity dependence on the cholesterol mole

fraction is linear throughout all experimentally

achiev-able mole fractions [40] Thus, the true maximal rate for

cholesterol in membranes has never been measured

Because microbial ChOxs are active with various

natural sterols, it is necessary to test the mole fraction

dependence using a more extensive variety of

mem-branes, composed of a variety of sterols, to determine

which substrate is the best

Moreover, the kcat*⁄ Km* varies depending on the

multibody interactions in the membrane When the

mole fraction of cholesterol exceeds a sustainable

packing ratio [6,19–21], the excess cholesterol is a

better substrate [7,25,40,43] Restated, cholesterol can

exist in membranes as free cholesterol clusters These

clusters appear above cholesterol⁄ phospholipid ratios

that depend on the precise lipid and method of

prepar-ing the membranes However, they are typically

detected above stoichiometries of 1⁄ 2 or 1 ⁄ 1 [6] All of

the results in these studies are consistent with a

catalytic model in which ChOx sits on the surface of

the membrane and binds sterol by passive partitioning

from the membrane into the active site

Thus, the question arises, what is the relevant

cholesterol matrix mixture to study? If the desired

use of ChOx is a technical application (e.g serum

cholesterol assays in the clinic), then micelles or

other non-native mixtures of cholesterol are the best

form of substrate to use in order to obtain maximal

activity By contrast, if the aim is to understand

physiological function, then cholesterol must be

assayed with lipid mixtures that reflect the native

environment of the enzyme There are three major

physiological functions of ChOxs that have been

studied to varying degrees The first function is in a

catabolic pathway for nutrition and the second

func-tion is a proposed role in virulence More recently,

ChOx has been implicated as a biosensor for

macro-lide biosynthesis

Cholesterol as a nutritional source

Conversion of a 3b-hydroxy-5-ene steroid to the corre-sponding 4-en-3-one product is the first and compul-sory step in bacterial sterol catabolic pathways Following this step, sterol-catabolizing microorganisms proceed to degrade the steroid nucleus and the sterol side chain simultaneously, but independently, at differ-ent rates Some species cleave the side chain before C-1(2) dehydrogenation and⁄ or 9a-hydroxylation of the steroid skeleton (Scheme 2, reviewed in [44]) More-over, an enzyme requiring O2 (ChOx) is always involved in the sterol 3b-hydroxy-5-ene conversion by actinomycete genera, Corynebacterium, Gordona, Proactinomyces and Rhodococcus; this conversion is carried out by a dehydrogenase⁄ isomerase enzyme that utilizes NAD+ or NADP+in a Pseudomonas sp [45], Comomonas testosteroni (formerly Pseudomonas testos-teroni) [46], Nocardia [47,48] and proteobacteria [49,50]

In mycobacteria, the existing evidence suggests that this step is catalyzed by a dehydrogenase [51], although an oxidase has been suggested to perform this function [52] (vide infra) Species of the genera Rhodococcus, Mycobacterium and Gordona are widespread in nature, where they play major roles in the degradation of organic waste, which includes sterols, and thus they have evolved the ability to use sterols as sources of carbon and energy Therefore, in the ChOx-producing species, one role of this enzyme is nutritional The precise matrix in which the substrate is presented to the enzyme is unclear In nature, phytosterols are common

in wood pulp waste streams and thus are likely to be found in membranous form Similarly, cholesterol, an animal product, is presumably presented as decaying membranous material to soil bacteria

Rhodococcal ChOx is an induced enzyme; its biosynthesis requires the presence of cholesterol or plant sterols (a detailed description of the level of production of ChOx in different strains is reported in the third review of this miniseries) ChOx induction in Rhodococcus sp GK1 is independent of the steroid 3b-hydroxy-5-ene, because cholest-4-en-3-one was demonstrated to be the inducer [53,54] Moreover, androstenedione or testosterone, intermediates in cholesterol catabolism (Scheme 2), completely repressed ChOx synthesis by this strain Thus, enzyme induction is dependent on the presence of the sterol side chain This regulation is consistent with the preferred substrates being cholesterol and phytosterols (e.g sitosterol and stigmasterol)

The exact taxonomy of some species of genera recognized to catabolize cholesterol has changed over time For example, Nocardia restrictus ATCC 14887, a

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strain extensively used by Sih et al [55,56] in their

studies of cholesterol catabolism, is now recognized as

R equi ATCC 14887 [57] A second example is the

ChOx-producing B sterolicum ATCC 21387, which is

actually an R equi strain [58] The original term,

nocardioform bacteria, encompasses the genera

Cory-nebacterium, Gordona (or Gordonia), Mycobacterium,

Nocardia, Rhodococcus and Tsukamurella Bacteria

belonging to these genera all contain mycolic acid in

their capsule and they are known as

mycolate-contain-ing nocardioform actinomycetes The term

nocardio-form describes the morphology and refers to mycelial

growth with fragmentation into rod-shaped and⁄ or

coccoid elements These actinomycetes form a distinct

suprageneric group; they are ubiquitous in nature and

have the ability to catabolize different natural

substances, including cholesterol and plant sterols The

group encompasses pathogenic species that are

gener-ally opportunistic New species are still being found,

for example, a Gordona actinomycete was recently isolated from sewage sludge and found to catabolize cholesterol [59]

Recent interest has focused on cholesterol oxidation

in mycobacteria There are many reports that myco-bacteria oxidize sterols [60–65] However, there is no definitive evidence that mycobacteria produce a ChOx The taxonomy of Mycobacterium sp used in the stud-ies of Stadtman and collaborators [66,67] has been revised, first to Nocardia cholesterolicum and finally to Rhodococcus rhodochrous [68,69] The enzyme isolated

by Stadtman et al [66] was a ChOx [68] Sequencing

of whole genomes has allowed a bioinformatics approach to gene identification No orthologs of the biochemically verified streptomycete (choA) and rhodo-coccal (choE) ChOx genes are present However, a putative ChOx was identified in the mycobacterial genomes and annotated as choD The choD gene is also present in the R equi genome [70] Phylogenetic

HO

cholesterol, 1

O

4-en-3-one, 2

HO2C

O

HO

O O

KstD

ChoE or Hsd

HO

O O

HO HsaA

O

O O

HO

CO2H

HO2C

HO2C

O

HsaC

propionyl CoA

acetyl CoA

HsaD

cyt P450

KshA KshB

O

O

HO2C

O OH

O

CO2H +

HO

CO2H HsaEFG

O

O

OH

dien-3-one, 3

6 7

8

9 10

11

12

13

KstD

oxygenase

fadD

acylCoA

synthetase

acylCoA

dehydrogenase

enoylCoA

dehyratase

O

SCoA CoAS

+

O

CoAS

side-chain β-oxidation

Cholesterol Metabolism

β-oxidation

β-oxidation

3-hydroxy-acylCoA

dehydrogenase

thiolase

O

CoAS

O

CoAS

O

CoAS

O

fadE

echA

fadA

OH

O

Scheme 2 Canonical cholesterol catabolism pathway [44] based on studies in Rhodococcus [100], Comamonas testosteroni [46,101] and fast-growing mycobacteria [102] It is believed that the cholesterol derivatives 2 and 3 are both substrates for the C26 hydroxylation enzyme The cholesterol side chain b-oxidation intermediates are potential substrates for KstD and KshA ⁄ B The preferred substrates have not been established.

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analysis of streptomycete, rhodococcal and

mycobacte-rial annotated ChOxs reveals that R equi ChoE is

56% identical to the ChOxs from Streptomyces species

(Fig 2 and [71]) The R equi and Streptomyces

proteins have signal peptide sequences, and their

corre-sponding ChOxs are, in most cases, extracellular

Although Rhodococcus is genetically more closely

related to Mycobacterium than to Streptomyces, the

identity of ChoE with the putative cholesterol oxidase

ChoD of Mycobacterium tuberculosis or

Mycobacte-rium leprae is low, around 25% In addition, ChoD

lacks a signal peptide sequence, suggesting that it is

localized inside the bacterium Importantly, M

tuber-culosis has a 3b-hydroxysteroid dehydrogenase

(Rv1106c, hsd) that has been expressed and purified

This enzyme converts NAD+ and cholesterol to

NADH and cholest-4-en-3-one [51] Disruption of the

hsd gene in M tuberculosis abrogates cholesterol

con-version to cholest-4-en-3-one, as determined by HPLC

analysis There is a report that expression of ChoD in

Mycobacterium smegmatis lysates increases cholesterol

oxidation activity [52] However, this activity was

measured colorimetrically and conversion of

choles-terol to cholest-4-en-3-one was not verified Taking

into consideration these observations, we support the

opinion of Navas and collaborators [71] who suggested

that ChoDs may be proteins without ChOx activity,

but belonging to the GMC oxidoreductase group

Cholesterol oxidase and virulence

Related to the high activity of ChOx with membranes

containing clusters of free cholesterol, treatment of cell

membranes with sphingomyelinase before the addition

of ChOx results in higher activity of the enzyme [72]

This activity is commonly reported as hemolysis

because red blood cells are used as the source of cell membranes This effect is seen both upon the addition

of purified enzymes, or upon the addition of bacterial strains that secrete sphingomyelin-specific phospholi-pase D and ChOx The hemolysis activity of R equi ChOx was confirmed through molecular genetic experi-ments [71] ChoE-negative R equi mutants lose cooper-ative hemolysis (CAMP reaction) that occurs with sphingomyelinase-producing Listeria ivanovii The CAMP reaction was also observed for Listeria mono-cytogenes and R equi [73] In other cases, the hydro-lases may be secreted by the same strain (e.g choline phosphohydrolase and sphingomyelinase C are pro-duced by R equi, which also produces ChOx) [74] These observations are consistent with a model in which ceramide formation displaces cholesterol from liquid-ordered regions [75] making the cholesterol more accessible to ChOx

These studies defined the parameters required for cell lysis and suggested a possible role for ChOx in pathogenesis All ChOxs isolated to date are extra-cellular, either secreted or cell associated R equi

is primarily a horse pathogen However, it is an emerging, opportunistic human infection, especially in immunocompromised individuals, for example, those infected with HIV [76] These bacteria infect and multiply inside the macrophage, a potentially rich source of cholesterol

An in vitro study suggested that during bacterial invasion of the host cell, membrane lysis is facilitated

by the induction of extracellular ChOx [77] The oxida-tion of macrophage membrane cholesterol by R equi (ATCC 33701) was studied under infection-mimicking conditions [78] In this study, the uptake of R equi cells by cultured mouse macrophages (ATCC PD388D1) was accompanied by intracellular survival

ChoE, R equi

ChoD, M marinum

C jeikeium

ChoD, M tuberculosis

PimE, S natalensis RimD, S diastaticus ChoA, Streptomyces sp

PteG, S avermitilis

ChoL, S virginiae

ChoD, R equi ChoD, M leprae

Fig 2 Unrooted phylogenetic tree for functionally characterized and putative cholesterol oxidase protein sequences from Streptomyces, Rho-dococcus and Mycobacterium The length of the horizontal lines corresponds to the relative evolutionary distance The tree was generated using a CLUSTALW2 alignment [104] with the neighbor-joining method [105] Proteins are identified by GenBank ID and gene ID if assigned: ORF1948, Rhodococcus equi ChoD; CAC44897, R equi ChoE; CAI36788, Corynebacterium jeikeium; CAR70482, Mycobacterium leprae ChoD; CAB01014, Mycobacterium tuberculosis H37Rv ChoD; ACC39597, Mycobacterium marinum ChoD; CAC20926, Streptomyces natalen-sis PimE; AAR16516, Streptomyces diastaticus RimD; ABS32193, Streptomyces virginiae ChoL; BAB69314, Streptomyces avermitilis PteG; AAA26719, Streptomyces sp ChoA.

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of the bacterium and enzymatic oxidation of

macro-phage cholesterol Cholesterol oxidation was

signifi-cantly increased when the strain was co-phagocytosed

with Corynebacterium pseudotuberculosis, a

sphingomy-elinase-producing bacterium and a cooperative partner

of R equi in the in vitro hemolysis of sheep

erythro-cytes [72] Synergistic actions of cytotoxic enzymes

may also take place in vivo, because pathogens and⁄ or

ubiquitous commensal organisms can exist at the same

time in infected hosts, especially in

immunocompro-mised individuals Moreover, intracellular survival of

the bacterium in the host macrophage is enhanced by

induction of the oxidative enzymes catalase (EC

1.11.1.6) and superoxide dismutase (EC 1.15.1.1) [77]

Both enzymes reduce oxidizing agents, hydrogen

per-oxide and free radicals and thus contribute to

patho-gen protection from oxidative stress effects These

studies did not address whether it is the lytic function

or the nutritional function that contributes to bacterial

survival in the macrophage

A mutant of R equi, originally isolated from foals

with pneumonia, in which choE (ChOx) was disrupted,

was constructed by allelic exchange This mutant was

devoid of ChOx activity The mutant was assessed for

in vivovirulence in mice or foals and for in vitro

cyto-toxicity to macrophages [79,80] The virulence of the

mutant strain was not attenuated and mutation did

not reduce cytotoxicity in infected macrophages Based

on the rates of multiplication of the mutant and the

parent strain in the infected animals or in the

phages, and their similar cytotoxicities to

macro-phages, it was concluded that ChOx is not a virulence

factor and its role may be limited to the catabolism of

cholesterol as a carbon and energy source of the

infect-ing bacterium Furthermore, a partial deletion mutant

in the supAB genes, which encode the permease

sub-units of the cholesterol uptake transporter (mce4) [81],

was used to infect macrophages in vitro and the

mutant was tested for growth on cholesterol as a sole

carbon source [82] Although disruption of the

per-mease genes blocks cholesterol catabolism, cholesterol

uptake and catabolism are not essential for survival of

R equiin the macrophage We note that in Rhodococcus,

ChOx is either secreted and⁄ or cell-surface-linked (see,

for example, [54,83–85]) Thus, the conversion of

cholesterol into cholest-4-en-3-one occurs outside the

bacterial cell, and the supAB⁄ mce4 transporter system

in rhodococci probably transports cholest-4-en-3-one

rather than cholesterol

These studies were all performed for short time

courses (2–4 weeks in foals) and it is possible that the

catabolism of cholesterol and⁄ or cell lysis mediated by

ChOx may be required at advanced stages of infection

Consistent with a catabolic role, the orthologous cho-lesterol transporter in M tuberculosis is required for bacterial persistence at the chronic stage of mouse lung infection, but not in the initial stages of infection [86]

In addition, the transporter is only required for growth within interferon-c-activated macrophages and its mutation has no effect on infection of resting macro-phages Both mycobacteria and rhodococci catabolize cholesterol, and the pathways share many similarities [87–89] However, in these two genera, the conversion

of cholesterol to cholest-4-en-3-one is catalyzed by dif-ferent enzymes – a dehydrogenase [51] and an oxidase – which are intracellular and extracellular (secreted and⁄ or cell-surface bound), respectively These differ-ences may be a consequence of additional functions that are distinct in the two genera The primary ques-tion, at this point in time, is whether cholesterol oxida-tion plays only a nutrioxida-tional role in pathogenesis, or if

it has additional consequences in microbial infection

In the host, cholesterol mixed with phospholipid or sphingomyelin is the presumed form of the substrate

in vivo and is the matrix that should be studied in assessments of cholesterol oxidation in pathogenesis

Cholesterol oxidase and polyene macrolide biosynthesis

Streptomyces natalensis produces the polyene macro-lide, pimaricin This macrolide is used in the food industry to prevent mold contamination of cheese and nonsterile food, and also for treatment of keratitis The mechanism of pimaricin antifungal activity relies

on its interaction with sterols, primarily ergosterol, in the cell membrane of molds, thus causing alteration of the membranes and the lysis of mold cells

Aparicio and collaborators [90,91] identified a gene cluster involved in pimaricin biosynthesis In the center

of this gene cluster is a gene, pimE, which encodes a cholesterol oxidase A transcriptional activator gene, pimR, is located at the 5¢-end of the cluster Disruption

of pimR results in total abrogation of pimE tion as well as a significant reduction in the transcrip-tion of biosynthetic genes, thus completely blocking the production of pimaricin [92]

PimE shares high amino acid identity with other known ChOxs that are in the GMC oxidoreductase family, including the active-site residues, and the enzyme is a catalytically active ChOx [93] The loca-tion of pimE in the middle of the pimaricin gene clus-ter is intriguing because the biosynthesis of this macrolide does not require cholesterol oxidation Moreover, the pimE gene is required for the produc-tion of pimaricin by S natalensis [93]

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Complementa-tion of the DpimE mutant restores macrolide

produc-tion Unexpectedly, when purified enzyme is added to

the growth media of the DpimE mutant, pimaricin

pro-duction is recovered ChOxs from other microbial

sources also restore pimaricin production in the DpimE

mutant, provided that they belong to the GMC

oxido-reductase family [i.e are type I cholesterol oxidases

(see the first review of this miniseries)] It is

hypothe-sized that the S natalensis ChOx acts as a signaling

protein for the macrolide biosynthesis pathway [94,95]

The regulatory model of pimaricin biosynthesis in

S natalensis cells is an attractive paradigm because

ChOx genes are present in additional antifungal

poly-ketide biosynthetic gene clusters in Streptomyces

[96,97] The precise mechanism of signaling is unclear

One possible mechanism is that ChOx acts as a fungal

sensor via oxidation of ergosterol, or another unknown

mold sterol Alternatively, the enzyme itself may act as

a ligand for a receptor signaling system

Early studies with extracellular ChOxs concluded

that ergosterol is a poor substrate for the enzyme from

Streptomyces [98] or from Rhodococcus sp [85]

However, to our knowledge, substrate specificity

studies with ergosterol have only been performed with

detergent micelles For these studies, the relevant form

of the substrate is ergosterol mixed with fungal lipids

Alternatively, the ergosterol may bind to the enzyme

without undergoing oxidation and induce a

conforma-tional change in PimE This activated complex would

then interact with a receptor signaling system to

promote pimaricin biosynthesis

Concluding comments

The present minireview considers the interaction of

microbial ChOx with biological membrane surfaces In

order to sequester its substrate, the ChOx molecule

binds to a membrane surface through hydrophobic

interactions However, the exact mechanisms of

bind-ing, sterol sequestration and 4-en-3-one release are not

well understood The questions that remain for future

investigation are: does the contact surface extend

beyond the entrance to the substrate-binding site, or

does any part of the protein insert more deeply than

another? In its interfacial mechanism, ChOx is similar

to the well-studied family of phospholipases

From the results of the studies analyzing the action

of ChOx on model membranes (both monolayers and

bilayers), it is known that binding and substrate

sequestration are the parameters that limit enzyme

activity Kinetically, the rate dependence on the

sub-strate mole fraction (k*cat⁄ K*m, Scheme 1), rather than

the bulk substrate concentration, is the important rate

constant to consider for determining substrate speci-ficity

The interfacial characteristics of ChOx are linked to its physiological roles as the enzyme that initiates ste-rol catabolism mainly in species of the actinomycetal genera, Corynebacterium, Gordona and Rhodococcus However, in the case of Mycobacterium and Nocardia, sterol conversion to the corresponding 4-en-3-one may

be carried out by a dehydrogenase⁄ isomerase system requiring NAD+or NADP+ Future studies that con-sider taxonomically well-determined mycobacterial members are needed to understand the role of choles-terol oxidation in these microorganisms

The actinomycetes encompass species that are gener-ally opportunistic pathogens A possible role of ChOx

in rhodococcal virulence has been proposed to be a consequence of the enzyme’s membrane-disruption characteristics, which were determined using model membranes and erythrocyte and macrophage cells The question of whether or not ChOx plays a role in virulence remains unanswered

The third possible function of ChOx (PimE) is as

a possible regulator in pimaricin biosynthesis by

S natalensis ChOx may act as a signaling protein via catalysis of mold ergosterol and⁄ or other sterols, or the enzyme itself may act as a ligand for a receptor signaling system, because PimE is extracellular [99] The receptor activator might be a PimE reaction product, or PimE itself could play the role of an acti-vating ligand In either case, elucidation of the precise mechanism by which ChOx promotes production of this macrolide is an interesting new avenue of research for an old enzyme

Acknowledgements

The work in the authors’ laboratories was supported

by the National Institutes of Health (AI065251, HL53306, N.S.S), the American Heart Association (0725861T, N.S.S.) and NATO (Collaborative Linkage Grant LST.CLG.980121)

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