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In this minireview, the main technologies used in metabonomics are summarized, brief details of the types of samples used are given, and the current phar-maceutical applications of metab

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Metabonomics in pharmaceutical R & D

John C Lindon, Elaine Holmes and Jeremy K Nicholson

Biomolecular Medicine, Faculty of Medicine, Sir Alexander Fleming Building, Imperial College London, UK

Introduction

Metabonomics has been formally defined [1] and can

be understood as the comprehensive and simultaneous

systematic determination of metabolite levels in whole

organisms and their changes over time as a

conse-quence of stimuli such as diet, lifestyle, environment,

genetic effects, and pharmaceutical interventions, both

beneficial and adverse For mammalian systems, this

involves the analysis of biofluids and tissues, and the

complex datasets are usually interpreted using

chemo-metric techniques [1,2] The approach builds on

meta-bolic analyses using NMR spectroscopy [3,4] and mass

spectrometry [5] first reported around 20 years ago,

and indeed it goes back to the concept suggested by Pauling et al in 1971 [6]

In this minireview, the main technologies used in metabonomics are summarized, brief details of the types of samples used are given, and the current phar-maceutical applications of metabonomics are des-cribed, but the important aspect of the measurement

of metabolic fluxes using stable isotope labeling is not covered here Some prospects for the future are dis-cussed later

Metabonomics studies of pharmaceutical relevance generally use biofluids, or cell or tissue extracts Urine and plasma are easily obtained, essentially

noninvasive-ly, and hence can be readily used for disease diagnosis

Keywords

biomarkers; diagnostics; drug safety;

metabonomics; spectroscopy

Correspondence

J C Lindon, Biomolecular Medicine, Faculty

of Medicine, Biomedical Sciences Division,

Imperial College London, Sir Alexander

Fleming Building, South Kensington,

London, SW7 2AZ, UK

Fax: +44 20 75 943 066

Tel: +44 20 75 943 194

E-mail: j.lindon@imperial.ac.uk

(Received 20 October 2006, revised 20

November 2006, accepted 30 November

2006)

doi:10.1111/j.1742-4658.2007.05673.x

This minireview is based on a lecture given at the First Maga Circe Confer-ence on metabolomics held at Sabaudia, Italy, in March 2006 in which the analytical and statistical techniques used in metabonomics, efforts at stan-dardization and some of the major applications to pharmaceutical research and development are reviewed Metabonomics involves the determination

of multiple metabolites simultaneously in biofluids, tissues and tissue extracts Applications to preclinical drug safety studies are illustrated by the Consortium for Metabonomic Toxicology, a collaboration involving several major pharmaceutical companies This consortium was able, through the measurement of a dataset of NMR spectra of rodent urine and serum samples, to build a predictive expert system for liver and kidney toxicity A secondary benefit was the elucidation of the endogenous bio-chemicals responsible for the classification The use of metabonomics in disease diagnosis and therapy monitoring is discussed with an exemplifica-tion from coronary artery disease, and the concept of pharmaco-meta-bonomics as a way of predicting an individual’s response to treatment is exemplified Finally, some advantages and perceived difficulties of the metabonomics approach are summarized

Abbreviations

CE, capillary electrophoresis; CLOUDS, classification of unknowns by density superposition; COSY, correlation spectroscopy; CPMG, Carr–Purcell–Meiboom–Gill; CSF, cerebrospinal fluid; DA, discriminant analysis; FT, Fourier transform; IBS, irritable bowel syndrome; LC-PUFA, long chain polyunsaturated fatty acid; MAS, magic angle spinning; OSC, orthogonal signal correction; PCA, principal component analysis; PLS, partial least squares; QC, quality control; SHY, statistical hetero-spectroscopy; STOCSY, statistical total correlation

spectroscopy; TOF, time of flight; TSP, trimethylsilylpropionic acid sodium salt; UPLC, ultra performance liquid chromatography.

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and in a clinical trials setting for monitoring drug

ther-apy However, there is a wide range of fluids that has

been studied, including seminal fluid, amniotic fluid,

cerebrospinal fluid, synovial fluid, digestive fluids,

blis-ter and cyst fluids, lung aspirates and dialysis fluids In

addition, a number of metabonomics studies have used

analysis of intact tissue biopsy samples and their lipid

and aqueous extracts [7] This particular approach can

also be used to characterize in vitro cell systems such as

tumor cells [8] and tissue spheroids [9]

Metabonomics analytical technologies

The principal analytical techniques that are employed

for metabonomic studies are based on NMR

spectro-scopy and mass spectrometry (MS) MS requires a

separation of the metabolic components using either

gas chromatography (GC) after chemical

derivatiza-tion, or liquid chromatography (LC), with the newer

method of ultra performance

increasingly Some users have advocated direct

injec-tion MS especially with the use of Fourier transform

mass spectrometers The use of capillary

electrophor-esis (CE) coupled to MS has also shown some

prom-ise Other more specialized techniques such as Fourier

transform infra-red (FTIR) spectroscopy and arrayed

electrochemical detection have been used in some cases

[10,11] The main limitation of these is the low level of

detailed molecular identification that can be achieved

However, the combination of retention time and redox

properties can serve as a basis for database searching

of libraries of standard compounds and the separation

output can also be directed to a mass spectrometer for

additional identification experiments

All metabonomics studies result in complex

multiva-riate datasets that require visualization software and

chemometric and bioinformatic methods for

interpret-ation The aim of these procedures is to produce

bio-chemically based fingerprints that are of diagnostic or

other classification value A second stage, crucial in

such studies, is to identify the substances causing the

diagnosis or classification, and these become the

com-bination of biomarkers that define the biological or

clinical context

NMR spectroscopy

Standard commercial NMR spectrometers can be

used for metabonomics, and for large scale

pharma-ceutical studies, automatic sample preparation is often

employed This can involve addition of buffer to

sta-bilize the pH, and D2O as a magnetic field lock signal

for the spectrometer NMR spectra typically take only

around 5 min to acquire using robotic flow-injection methods For large scale studies, barcoded vials con-taining the biofluid can be used and the contents of these can be transferred and prepared for analysis using robotic liquid handling technology into 96-well plates under laboratory information management sys-tem control Alternatively, for more precious samples

or for those of limited volume, conventional 5 mm or capillary NMR tubes are usually used, either individu-ally or using a commercial sample tube changer and automatic data acquisition

The large interfering NMR signal arising from water

in all biofluids is eliminated by use of standard NMR solvent suppression pulse sequences The reference compound used in aqueous media is usually the sodium salt of 3-trimethylsilylpropionic acid (TSP), with the methylene groups deuterated to avoid giving rise to peaks in the 1H NMR spectrum Absolute con-centrations can be obtained if the sample contains an added internal standard of known concentration, or if

a standard addition of the analyte of interest is added

to the sample, or if the concentration of a substance is known by independent means (e.g., many metabolites can be quantified by conventional biochemical assays) Whilst a1H NMR spectrum of urine contains thou-sands of sharp lines from predominantly low molecular mass metabolites, blood plasma and serum contain both low and high molecular mass components, and these give a wide range of signal line widths Broad bands from protein and lipoprotein signals contribute strongly to the 1H NMR spectra, with sharp peaks from small molecules superimposed on them [12] Standard NMR pulse sequences, where the observed peak intensities are edited on the basis of molecular diffusion coefficients or on NMR relaxation times, can

be used to select only the contributions from macro-molecules, or alternatively to select only the signals from the small molecule metabolites, respectively It is also possible to use these approaches to investigate molecular mobility and flexibility, and to study inter-molecular interactions such as the reversible binding between small molecules and proteins

The development of high resolution1H magic angle spinning (MAS) NMR spectroscopy has allowed the acquisition of high resolution NMR data on small pieces of intact tissues with no pretreatment [7,13] Rapid spinning of the sample (typically at  4–6 kHz)

at an angle of 54.7 relative to the applied magnetic field serves to reduce the loss of information caused by line broadening effects seen in nonliquid samples such

as tissues MAS NMR spectroscopy has straightfor-ward, but manual, sample preparation NMR spectro-scopy on a tissue sample in an MAS experiment is the

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same as solution state NMR and all common pulse

techniques can be employed in order to study

meta-bolic changes and to perform molecular structure

elu-cidation and molecular dynamics studies

Some typical 1H NMR spectra are given in Fig 1

showing the different profiles from rat liver tissue

(using MAS NMR spectroscopy), urine and blood

plasma

Two-dimensional NMR spectroscopy can be useful

for increasing signal dispersion and for elucidating

the connectivities between signals, thereby aiding

bio-marker identification Those of principal use include

1H-1H 2D J-resolved spectroscopy, which attenuates

the peaks from macromolecules and yields information

on the multiplicity and coupling patterns of resonances

and 1H-1H spin connectivity experiments known as

correlation spectroscopy (COSY) and total correlation

spectroscopy (TOCSY), giving information on which

hydrogens in a molecule are close in chemical bond

terms Use of other types of nuclei, such as naturally

abundant 13C or 15N, or where present 31P, through

heteronuclear correlation experiments, can be import-ant to help assign NMR peaks These experiments benefit from the use of so-called inverse detection, where the lower sensitivity or less abundant nucleus NMR spectrum (such as 13C) is detected indirectly using the more sensitive⁄ abundant nucleus (1H) The commercialization of cryogenic probes where the detector coil and preamplifier (but not the samples) are cooled to around 20K is already proving useful for metabonomics studies This has provided an improve-ment in spectral signal⁄ noise ratios of up to a factor of five by reducing the thermal noise in the electronics of the spectrometer Conversely, a reduction in data acquisition times by up to a factor of 25 become poss-ible for the same amount of sample NMR spectro-scopy of biofluids detecting the much less sensitive13C nuclei which also only have a natural abundance (1.1%) also becomes possible because of the increase

in signal-to-noise ratio [14] This technology also makes the use of tissue-specific microdialysis samples more feasible [15]

A

B

7.5 7.0 6.5 6.0 5.5 5.0 4.5 4.0 3.5 3.0 2.5 2.0 1.5 δ 1H

5.5 6.0 8.2

C

Fig 1 (A) 600 MHz standard solvent sup-pression pulse 1 H NMR spectrum of rat urine (B) 600 MHz Carr–Purcell–Meiboom– Gill (CPMG) 1 H NMR spectrum of rat blood plasma (C) 600 MHz CPMG 1 H MAS NMR spectrum of the left lateral lobe of a rat liver.

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Mass spectrometry

Mass spectrometry coupled to a chromatographic

separ-ation has also been widely used in metabolic

fingerprint-ing and metabolite identification Although most studies

to date have been on plant extracts and model cell

sys-tem extracts [16], its application to mammalian studies

is increasing MS is inherently considerably more

sensi-tive than NMR spectroscopy provided the metabolite

ionizes, but it is generally necessary to employ different

separation techniques for different classes of substances

Analyte quantitation by MS in complex mixtures of

highly variable composition can be impaired by variable

ionization and ion suppression effects For plant

meta-bolic studies, most investigations have used chemical

derivatization to ensure volatility and analytical

repro-ducibility, followed by GC-MS analysis Some

approa-ches using MS rely on more targeted studies, for

example by detailed analysis of lipids [17]

For metabonomics applications on biofluids, an

HPLC chromatogram is generated with MS detection,

usually using electrospray ionization, and both positive

and negative ion chromatograms can be measured At

each sampling point in the chromatogram there is a

full mass spectrum and so the data is three-dimen-sional in nature, i.e., retention time, mass and inten-sity Given this very high resolution it is possible to cut out any mass peaks from interfering substances such as drug metabolites, essentially without affecting the structure of the dataset

The problem of ion suppression is minimized by improving the efficiency of the chromatography and this has been achieved using UPLC This is a combina-tion of a 1.7 lm reversed-phase packing material, and

a chromatographic system, operating at around 827.4 bar

2 UPLC provides around a 10-fold increase in speed and a three- to five-fold increase in sensitivity compared to a conventional stationary phase

UPLC-MS has already been used for metabolic profiling of urines in a number of rodent studies [18] A compar-ison of data generated using both HPLC-MS and UPLC-MS is given in Fig 2

CE coupled to mass spectrometry has also been explored as a possible technology for metabonomics studies [19] Metabolites are first separated by CE based on their charge and size and then selectively detected using MS, and the technique has been applied

to studies of bacterial growth

HPLC

UPLC

6 8 7.5 10

150 300 450

600 750 m/z 150 300 450 600 750 m/z

9000

7500

6000

4500

3000

1500

0 cm

1750

1500

1000 1250

750

500

250

0 cm

Fig 2 Three-dimensional plots of retention time, m ⁄ z and intensity from control white male mouse urine using (left) HPLC-MS with a 2.1 cm · 100 mm Waters

5 Symmetry 3.5 lm C18 column (Milford, MA, USA), eluted with 0–95% linear gradient of water with 0.1% (v ⁄ v) formic acid:acetonitrile with 0.1% (v ⁄ v) formic acid over 10 min at a flow rate of 0.6 mLÆmin)1and (right) UPLC-MS with 2.1 cm · 100 mm Waters ACQUITY 1.7 lm C18 column, eluted with the same solvents at a flow rate of 0.5 mLÆmin)1 In both cases, the column eluent was monitored by ESI orthogonal acceleration

6 -TOF-MS from 50 to 850 m ⁄ z in positive ion mode Reproduced with permission from Wilson

et al [18].

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For biomarker identification, it is also possible to

separate out substances of interest on a larger scale

from a complex biofluid sample using techniques such

as solid phase extraction or HPLC For metabolite

identification, directly coupled

chromatography-spectro-scopy methods can also be used The most general of

these ‘hyphenated’ approaches is HPLC-NMR-MS [20]

in which the eluting HPLC peak is split, with parallel

analysis by directly coupled NMR and MS techniques

This can be operated in on-flow, stopped-flow and

loop-storage modes, and thus can provide the full

array of NMR and MS-based molecular identification

tools These include 2D NMR spectroscopy as well as

MS-MS for identification of fragment ions and Fourier

transform (FT)-MS or time of flight (TOF)-MS for

accurate mass measurement and hence derivation of

molecular empirical formulae

Chemometrics methods

One common objective in metabonomics is to classify a

sample based on identification of inherent patterns of

peaks in a dataset (usually a spectrum) and secondly to

identify those spectral features responsible for the

clas-sification The approach can also be used for reducing

the dimensionality of complex datasets, for example by

2D or 3D mapping procedures, to enable easy

visual-ization of any clustering or similarity of the various

samples Alternatively, in what are known as

‘super-vised’ methods, multiparametric datasets can be

mod-elled so that the class of separate samples (a ‘validation

set’) can be predicted based on a series of mathematical

models derived from the original data or ‘training set’

One popular technique that has been used

exten-sively in metabonomics is principal components

analy-sis (PCA) Each PC is a linear combination of the

original data parameters (e.g., intensity values for a

range of ion m⁄ z-values from MS) and each successive

PC explains the maximum amount of variance

poss-ible, not accounted for by the previous PCs Each PC

is orthogonal and therefore independent of the other

PCs and so the variation in the spectral set is usually

described by many fewer PCs than comprise the

num-ber of original data point values, because the less

important PCs describe the noise variation in the

spec-tra Conversion of the data to PCs results in two

mat-rices known as scores and loadings Scores, the linear

combinations of the original variables, can be regarded

as the new variables, and in a scores plot each point

represents a single sample spectrum The PC loadings,

where each point represents a different spectral

inten-sity, define the way in which the old spectral variables

are linearly combined to form the new variables and

show those variables carrying the greatest weight in determining the positions of the points in the scores plot In addition, there are many other visualization (or unsupervised) methods such as nonlinear mapping and hierarchical cluster analysis

To illustrate PCA, Fig 3 shows the scores and loa-dings plots for PC1 versus PC2 for data from a series

of1H NMR spectra of rat urine in a toxicity study In

Fig 3 Results of a principal components analysis based on NMR spectra of urine from rats treated with control dosing vehicle, or one of the two liver toxins, a-naphthylthioisocyanate (ANIT) or hydrazine (A) PC scores plot (PC1 versus PC2) where each point corresponds to a single urine sample, showing clear clustering of the samples from control urine and from the toxin-treated animals The liver toxins form separate clusters because they have different biochemical mechanisms of action and hence different biochemical profiles in the urine (B) The corresponding PC loadings plot where each point corresponds to a specific NMR spectral region, leading

to the possibility of identifying biomarkers of the toxicological effect For example, in the scores plot, the points corresponding to urines from hydrazine-treated animals appear in the lower left quad-rant and in the corresponding loadings plot, this region indicated that NMR peaks from 2-AA (2-aminoadipate) were important, and thus this is a biomarker of the hydrazine-induced toxicity.

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the scores plot, each point represents a single NMR

spectrum and the clustering of points shows the

differ-ent biochemical effects of the two differdiffer-ent toxins

rel-ative to the control group In cases where samples are

collected over time, onset and recovery trajectories can

be observed The loadings plot indicates which regions

of the NMR spectra are responsible for the clustering

If a predictive model is required, one widely used

supervised method (i.e using a training set of data with

known outcomes) is projection on latent structures

(PLS) This is a method that relates a data matrix

con-taining variables from samples, such as spectral

inten-sity values (an X matrix), to a matrix containing

outcome variables (e.g., measurements of response, such

as toxicity scores) for those samples (a Y matrix) PLS

can also be used to examine the influence of time on a

dataset, which is particularly useful for biofluid NMR

data collected from samples taken over a time course of

the progression of a pathological effect PLS can also be

combined with discriminant analysis (DA) to establish

the optimal position to place a discriminant surface that

best separates classes It is important to build and test

such chemometric models using independent training

data and validation datasets Extensions of this

approach allow the evaluation of those descriptors that

are completely independent (orthogonal) to the Y

mat-rix of end-point data This orthogonal signal correction

(OSC) can be used to remove irrelevant and confusing

parameters and has been integrated into the PLS

algo-rithm [21] If the Y matrix contains continuous data,

then PLS regression is a very useful approach

There is a variety of other methods that use nonlinear

combinations of the data variables and these include

genetic algorithms, machine learning, Bayesian

mode-ling and artificial neural networks In these, a training

set of data is used to develop algorithms, which ‘learn’

the structure of the data and can cope with complex

functions For example, probabilistic neural networks

have shown promise for predicting toxicity from

NMR-based metabonomics data [22]

It should be emphasized that both unsupervised and

supervised approaches can be useful in metabonomics

Unsupervised methods provide information on the

nat-ural structure of the data, whilst for supervised

meth-ods it is vital to carry out proper validation of the

models generated, involving training datasets and blind

validation and test sets of data

Statistical spectroscopy for biomarker

identification

Recently, a new method for identifying multiple

NMR peaks from the same molecule in a complex

mixture, hence providing a new approach to molecu-lar identification, has been introduced This is based

on the concept of statistical total correlation spectro-scopy and has been termed STOCSY [23] This takes advantage of the colinearity of many of the intensity variables in a set of spectra (e.g., 1H NMR spectra)

so that a pseudo-2D NMR spectrum can be calcula-ted that displays the correlation among the intensities

of the various peaks across the whole sample This method is not limited to the usual connectivities that are deducible from more standard 2D NMR spectro-scopic methods, such as TOCSY Added information

is available by examining lower correlation coeffi-cients or even negative correlations because this leads

to connection between two or more molecules involved in the same biochemical pathway In an extension of the method, the combination of STO-CSY with supervised chemometrics methods offers a new framework for analysis of metabonomic data In

a first step, a supervised multivariate discriminant analysis can be used to extract the parts of NMR spectra related to discrimination between two sample classes This information is then combined with the STOCSY results to help identify the molecules responsible for the metabolic variation The applica-bility of the method is illustrated in Fig 4, where a spin system of two triplets can be noticed at d 2.91 and d 2.51 This spin system is strongly correlated to others resonances in the aromatic region of the spec-trum, although not spin-coupled Computing only the correlation between one of the data points represent-ing the maximum of one of the triplets, and all the other variables leads to a single vector, which has the size of the number of variables used Then, by selecting the spectrum with the maximum value of this selected variable, it is possible to plot it with a colour code corresponding to the correlation between the selected resonance and all the other points of the spectra Correlations can be observed between reso-nances with no NMR-based spin-coupling connectiv-ity Thus, in the aromatic region, shown in Fig 4, it

is possible to recognize the resonances of a meta-substituted benzene ring (one triplet, two doublets, and one singlet) Thus, this molecule can be identi-fied as a derivative of a meta-substituted phenylprop-anoic acid and is probably 3-hydroxyphenylpropionic acid

The approach is not limited to NMR spectra alone and has been extended to other forms of data It has recently been applied to coanalysis of both NMR and mass spectra from a metabonomic toxicity study [24] This allowed better assignment of biomarkers of the toxin effect by using the correlated but complementary

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information available from the NMR and mass spectra

taken on a whole sample cohort

Standards and reporting needs in metabonomics

Several initiatives have been under way to investigate

the reporting needs and standardization of reporting

arrangements for metabonomics studies The Standard

Metabolic Reporting Structures group (http://

www.smrsgroup.org) has produced a draft policy

document covering all of those aspects of a metabolic

study that are recommended for recording, from the

origin of a biological sample, the analysis of material

from that sample, and chemometric and statistical

approaches to retrieve information from the sample

data, and a summary publication has appeared [25]

The various levels and consequent detail for reporting

needs, including journal submissions, public databases

and regulatory submissions have also been addressed

In parallel, a scheme called ArMet for capturing data

and metadata from metabolic studies has been

pro-posed and developed [26] These activities were

fol-lowed up in August 2005 with a workshop and

discussion meeting sponsored by the US National

Institutes of Health, from which plans are being

devel-oped to define standards in a number of areas relevant

to metabonomics, including characterization of

sam-ple-related metadata, technical standards and related

data, metadata and quality control matters for the

analytical instrumentation, data transfer methodologies

and schema for implementation of such activities, and

development of standard vocabularies to enable

trans-parent exchange of data [27] For details of the current

activity in this area the reader is referred to the Meta-bolomics Standards Initiative (http://msi-workgroups sourceforge.net/)

Pharmaceutical R & D metabonomics applications

Physiological and gut microfloral effects

A good understanding of normal biochemical profiles

is a prerequisite for evaluation of metabolic changes caused by xenobiotics or disease Thus metabonomics has been used to identify metabolic differences, in experimental animals such as mice and rats, caused by

a range of inherent and external factors [28] These dif-ferences may help explain differential toxicity of drugs between strains and interanimal variation within a study Many effects can be distinguished, including male⁄ female differences, age-related changes, estrus cycle effects in females, diet, diurnal effects, differenti-ation of wildtype and genetically modified animals, and interspecies differences and similarities using both NMR- and MS-based approaches

The importance of the symbiotic relationship between mammals and their gut microfloral popula-tions has been recognized [29] and highlighted in sev-eral studies These include a study in which axenic (germ free) rats were allowed to recolonize their gut microflora in normal laboratory conditions with their urine biochemical profiles being monitored for 21 days using 1H NMR spectroscopy [30], and the combined effects of gut bacteria and gut parasites on metabolic profiles [31]

A

B

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7.4 7.3 7.2

HO

HO

O

1

0.95

0.85

0.75

0.65 0.7 0.8 0.9

δ 1 H (p.p.m.)

r 2

δ 1 H (p.p.m.)

2.8 2.7 2.6 2.5

Fig 4 One-dimensional STOCSY analysis to identify peaks correlated to that at the chemical shift, d 2.51 The degree of correla-tion across the spectrum has been colour-coded and projected on the spectrum (A) Full spectrum; (B) partial spectrum between d 7.1–7.5; (C) partial spectrum between d 2.4–3.0 The STOCSY procedure enabled the assignment of this metabolite

as 3-hydroxyphenylpropionic acid Adapted from Cloarec et al [23]

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The metabolic consequences of early life maternal

separation stress have been investigated in rats, either

alone or in combination with secondary acute water

avoidance stress [32] The effect of a long chain

poly-unsaturated fatty acid (LC-PUFA) enriched dietary

intervention (postulated to be beneficial) in stressed

animals was also assessed Systematic changes in

meta-bolic biochemistry were evaluated using 1H NMR

spectroscopy of blood plasma and multivariate pattern

recognition techniques The biochemical response to

stress was characterized by decreased levels of total

lipoproteins and increased levels of amino acids,

glucose, lactate, creatine and citrate Secondary acute

water avoidance stress also caused elevated levels of

O-acetyl glycoproteins in blood plasma LC-PUFA

dietary enrichment did not alter the metabolic response

to stress but did result in a modified lipoprotein

pro-file This work indicated that the different stressor

types resulted in some common systemic metabolic

responses that involve changes in energy and muscle

metabolism, but that they are not reversible by dietary

intervention

Irritable bowel syndrome (IBS) is a common

multi-factorial intestinal disorder for which the aetiology

remains largely undefined, and recently, using a

Trichi-nella spiralis-induced model of postinfective IBS, the

effects of probiotic bacteria on gut dysfunction have

been investigated using metabonomics [33] Mice were

divided into four groups: an uninfected control group

and three T spiralis-infected groups, one as infected

control and the two other groups subsequently treated

with either Lactobacillus paracasei or L paracasei-free

medium Plasma, jejunal wall and longitudinal

myen-teric muscle samples were collected at day 21

postinfec-tion, and NMR spectroscopy was used to characterize

these and plasma metabolic profiles T spiralis-infected

mice showed an increased energy metabolism, fat

mobilization and a disruption of amino acid

meta-bolism due to increased protein breakdown, which

were related to the intestinal hypercontractility

Increased levels of taurine, creatine and

glycerophos-phorylcholine in the jejunal muscles were associated

with the muscular hypertrophy and disrupted jejunal

functions L paracasei treatment normalized the

mus-cular activity and the disturbed energy metabolism as

evidenced by decreased glycogenesis and elevated lipid

breakdown in comparison with untreated T

spiralis-infected mice Changes in the levels of plasma

metabo-lites (glutamine, lysine, methionine) that might relate

to a modulation of immunological responses were also

observed in the presence of the probiotic treatment

The work suggested that probiotics may be beneficial

in patients with IBS

Pre-clinical drug candidate safety assessment

In vivo preclinical drug safety assessment remains one

of the main bottle-necks in pharmaceutical R & D and

is a prime target for improving efficiency in drug development

Having defined the metabolic hyperspace occupied

by normal animals, metabonomics can be used for rapid classification of a biofluid sample as normal or abnormal Classification of the target organ or region

of toxicity, the biochemical mechanism of a toxin, the identification of combination biomarkers of toxic effect and evaluation of the time course of the effect, e.g., the onset, evolution and regression of toxicity, can all

be determined There have been many studies using

1H NMR spectroscopy of biofluids to characterize drug toxicity going back to the 1980s [3], and the role

of metabonomics in particular, and magnetic reson-ance in general in toxicological evaluation of drugs has been comprehensively reviewed [34] The situation is now changing with the introduction of the combined use of NMR spectroscopy and HPLC-MS for toxicity studies

The usefulness of NMR-based metabonomics for the evaluation of xenobiotic toxicity effects has recently been comprehensively explored by the successful Con-sortium for Metabonomic Toxicology This was con-ducted by five pharmaceutical companies and Imperial College, London, UK [35], and its aim was to develop methodologies for the acquisition of metabonomic data using 1H NMR spectroscopy of urine and blood serum from rats and mice for preclinical toxicological screening of candidate drugs, to build databases of spectra and to develop an expert system for predicting target organ toxicity

To assess the levels of analytical and biological vari-ation that could arise through the use of metabonom-ics on a multisite basis, a feasibility study was carried out at the start of the project, using the same detailed protocol and using the same model toxin, across all company sites The biological variability was evaluated

by a detailed comparison of the ability of the compan-ies to provide consistent urine and serum samples for

an in-life study of the same toxin There was a high degree of consistency between samples from the various companies and dose-related effects could be distinguished from intersite variation An intersite NMR analytical reproducibility study also revealed a high degree of robustness giving a multivariate coeffi-cient of regression between paired samples of only about 1.6% [36]

To achieve the project goals, new methodologies for analyzing and classifying the complex datasets were

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developed The predictive expert system that was

developed takes into account the metabolic trajectory

over time, and so a new way of comparing and scaling

these multivariate trajectories was developed [37] A

novel classification method for identifying the class of

toxicity based on all of the NMR data for a given

study was also developed This has been termed

‘Clas-sification Of Unknowns by Density Superposition

(CLOUDS)’ [38] and is a novel non-neural

implemen-tation of a classification technique developed from

probabilistic neural networks

This consortium showed that it is possible to

con-struct predictive and informative models of toxicity

using NMR-based metabonomic data, delineating the

whole time course of toxicity Curated databases of

spectral ( 35 000 NMR spectra) and conventional

(clinical chemistry, histopathology, etc.) results for 147

model toxins and treatments that served as the basis

for computer-based expert systems for toxicity

predic-tion were also produced All of the project goals

inclu-ding provision of multivariate statistical models (expert

systems) for prediction of toxicity, initially for liver

and kidney toxicity in the rat and mouse were

achieved, and the predictive systems and databases

were transferred to the sponsoring companies [39]

Clinical pharmaceutical applications

Many examples exist in the literature on the use of

NMR-based metabolic profiling to aid human disease

diagnosis, such as the investigation of diabetes using

plasma and urine, neurological conditions such as

Alzheimer’s disease using cerebrospinal fluid, arthritis

using synovial fluid and male infertility using seminal

fluid In addition, analysis of urine has been used in

the investigation of drug overdose, renal

transplanta-tion and various renal diseases NMR spectroscopy

of urine and plasma has been used extensively for

the diagnosis of inborn errors of metabolism in

chil-dren [40] Most of the earlier studies using NMR

spectroscopy have been reviewed previously [41], but

more recent studies include cerebrospinal fluid sample

analysis using NMR spectroscopy to distinguish

var-ious types of meningitis infection (bacterial, viral and

fungal) [42] and to investigate subarachnoid

haemor-rhage [43] Human serum samples have been analysed

using NMR spectroscopy to develop a diagnostic

method for coronary artery disease [44] One area of

disease where progress is being made using

NMR-based metabonomics studies of biofluids is cancer

This is highlighted by a publication on the diagnosis

of epithelial ovarian cancer based on analysis of

serum [45]

Pharmacometabonomics For personalized healthcare, an individual’s drug treat-ments must be tailored so as to achieve maximal effic-acy and avoid adverse drug reactions One of the approaches has been to understand the genetic

make-up of different individuals (pharmacogenomics) and to relate these to their varying abilities to handle pharma-ceuticals both for their beneficial effects and for identi-fying adverse effects Very recently, an alternative approach to understanding such intersubject variability has been developed using metabonomics, and used to predict the metabolism and toxicity of a dosed sub-stance, based solely on the analysis and modeling of a predose metabolic profile [46] Unlike pharmaco-genomics, this approach, which has been termed ‘phar-macometabonomics’, is sensitive to both the genetic and modifying environmental influences that determine the metabolic fingerprint of an individual This new approach has been illustrated with studies of the toxic-ity and metabolism of compounds with very different modes of action (allyl alcohol, galactosamine and acet-aminophen) administered to rats

Integration of -omics results

The value of obtaining multiple NMR spectroscopic and⁄ or LC-MS datasets from various biofluid samples and tissues of the same animals collected at different time points has been demonstrated This procedure has been termed ‘integrated metabonomics’ [2] and can be used to describe the changes in metabolism in different body compartments affected by exposure to, for exam-ple, toxic xenobiotics If profiles are obtained over time, they provide extra information and are character-istic of particular types and mechanisms of pathology Samples from multiple sources give a more complete description of the biochemical consequences than can

be obtained from one fluid or tissue alone

Although this review concentrates on metabolic ana-lyses, there is a requirement to integrate information at the transcriptomic, proteomic and metabonomic levels, despite these different levels of biological control showing very different time scales of change This is because some time courses can be very rapid, such as gene switching, some require much longer time scales, e.g protein synthesis, or in the case of metabolic chan-ges, can encompass enormous ranges of time scales Biochemical changes do not always occur in the order, transcriptomic, proteomic, metabolic, because, for example, pharmacological or toxicological effects at the metabolic level can induce subsequent adaptation effects at the proteomic or transcriptomic levels One

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important potential role for high-throughput and

highly automated metabonomics methods therefore

could be to direct the timing of more expensive or

labour-intensive proteomic and transcriptomic analyses

in order to maximize the probability of observing

meaningful and relevant biochemical changes using

those techniques

In addition, overlaid with this temporal complexity,

is the fact that environmental and lifestyle effects have

a large effect at all levels of molecular biology Gene

and protein expression effects and metabolite levels

can be altered by such factors, and this variation has

to be incorporated into any analysis as part of

inter-sample and interindividual variation Even healthy

ani-mals and man can be considered as ‘super-organisms’,

with an internal ecosystem of diverse symbiotic gut

microflora that have metabolic processes that interact

with the host and for which, in many cases, the

gen-ome is not known The complexity of mammalian

bio-logical systems and the diverse features that need to be

measured to allow ‘-omics’ data to be fully interpreted

have been reviewed recently [47] and it has been

argued that novel approaches will continue to be

required to measure and model metabolic processes in

various compartments from such global systems with

different interacting cell types, and with various

geno-mes, connected by cometabolic processes

Integration of metabonomics data with that from

other multivariate techniques in molecular biology

such as from gene array experiments or proteomics is

now becoming a reality Pharmaceutically related

examples include phenotypic differences [48] and

toxic-ity studies of acetaminophen [49], bromobenzene

[50,51], a-naphthylisothiocyanate [52], hydrazine [53]

and methapyriline [54]

Future promise

In summary, it is clear that metabonomics will have an

impact in pharmaceutical R & D but some potential

disadvantages of the approach include the use of

mul-tiple analytical technologies with different sensitivities,

dynamic ranges and metabolite detection abilities and

the complexity of the resulting datasets Through the

inappropriate application of chemometrics, it is

poss-ible to over-interpret the data, but this is easily

avoi-ded by correct statistical rigour There remains a need

for the regulatory agencies to be trained in the

inter-pretation of the data and for the availability of more

well trained practitioners generally

However, on the other hand, the analytical

proce-dures used are stable and robust, and have a high

degree of reproducibility, and although advances will

obviously be made in the future, current data will always be readable and interpretable In contrast to other -omics, metabonomics enjoys a good level of biological reproducibility and the cost per sample and per analyte is relatively low It has the advantage of not having to preselect analytes, and generally it is minimally invasive with hypothesis generation studies being easily possible Metabolic biomarkers are closely identifiable with real biological endpoints and provide

a global systems interpretation of biological effects, including the interactions between multiple genomes such as humans and their gut microflora One major potential strength of metabonomics is the possibility that metabolic biomarkers will be more easily used across species than transcriptomic or proteomic bio-markers and this should be important for pharmaceuti-cal studies

For complex disease and drug effect evaluation, combinations of biomarkers are likely to be necessary and thus there will be many opportunities for metabo-nomics that are as yet under-explored, such as its use

in environmental toxicity studies, its use in directing the timing of transcriptomic and proteomic experi-ments, and its use for deriving theranostic biomarkers

It will surely be an integral part of any multiomics study where all the datasets are combined in order to derive an optimum set of biomarkers

References

1 Nicholson JK, Lindon JC & Holmes E (1999) Metabo-nomics’: Understanding the metabolic responses of liv-ing systems to pathophysiological stimuli via

multivariate statistical analysis of biological NMR spectroscopic data Xenobiotica 29, 1181–1189

2 Nicholson JK, Connelly J, Lindon JC & Holmes E (2002) Metabonomics: a platform for studying drug toxicity and gene function Nat Rev Drug Disc 1, 153–162

3 Nicholson JK & Wilson ID (1989) High-resolution pro-ton magnetic resonance spectroscopy of biological fluids Prog NMR Spectrosc 21, 449–501

4 Gartland KPR, Beddell CR, Lindon JC & Nicholson

JK (1991) The application of pattern recognition meth-ods to the analysis and classification of toxicological data derived from proton NMR spectroscopy of urine Mol Pharmacol 39, 629–642

5 van der Greef J, Tas AC, Bouwman J, Ten Noever de Brauw MC & Schreurs WHP (1983) Evaluation of field-desorption and fast atom-bombardment mass spectro-metric profiles by pattern recognition techniques Anal Chim Acta 150, 45–52

6 Pauling L, Robinson AB, Teranishi R & Cary P (1971) Quantitative analysis of urine vapor and breath using

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