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Alternative binding proteins: Anticalins – harnessing the structural plasticity of the lipocalin ligand pocket to engineer novel binding activities Arne Skerra Lehrstuhl fu¨r Biologische

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Alternative binding proteins: Anticalins – harnessing the structural plasticity of the lipocalin ligand pocket to

engineer novel binding activities

Arne Skerra

Lehrstuhl fu¨r Biologische Chemie, Technische Universita¨t Mu¨nchen, Freising-Weihenstephan, Germany

Lipocalins occur in many organisms, such as

verte-brates, insects and plants, and even in bacteria, where

their physiological role usually lies in the transport or

storage of hydrophobic and⁄ or chemically sensitive

organic compounds, especially vitamins, lipids, steroids

and other secondary metabolites [1] Currently, the

number of assigned lipocalin sequences has grown

beyond 500 [2] and for more than 100 members of this

family the 3D structure has been described [3] In the

human body up to 12 different lipocalins, which exert diverse physiological functions, have been identified [4]: a1-acid glycoprotein, a1-microglobulin, apolipopro-tein D (ApoD), apolipoprotein M, complement component 8c, the epididymal retinoic acid-binding protein, glycodelin, neutrophil gelatinase-associated lipocalin (NGAL, Lcn2), odorant-binding protein, prostaglandin D synthase, retinol-binding protein and tear lipocalin (Tlc, Lcn1)

Keywords

bacterial expression; b-barrel; CTLA-4;

digitalis; fluorescein; ligand binding; lipocalin;

molecular recognition; protein engineering;

VEGF

Correspondence

A Skerra, Lehrstuhl fu¨r Biologische Chemie,

Technische Universita¨t Mu¨nchen, An der

Saatzucht 5, 85350 Freising-Weihenstephan,

Germany

Fax: +49 8161 714352

Tel: +49 8161 714351

E-mail: skerra@wzw.tum.de

(Received 16 November 2007, revised 9

March 2008, accepted 22 March 2008)

doi:10.1111/j.1742-4658.2008.06439.x

Antibodies are the paradigm for binding proteins, with their hypervariable loop region supported by a structurally rigid framework, thus providing the vast repertoire of antigen-binding sites in the immune system Lipoca-lins are another family of proteins that exhibit a binding site with high structural plasticity, which is composed of four peptide loops mounted on

a stable b-barrel scaffold Using site-directed random mutagenesis and selection via phage display against prescribed molecular targets, it is possi-ble to generate artificial lipocalins with novel ligand specificities, so-called anticalins Anticalins have been successfully selected both against small hapten-like compounds and against large protein antigens and they usually possess high target affinity and specificity Their structural analysis has yielded interesting insights into the phenomenon of molecular recognition Compared with antibodies, they are much smaller, have a simpler molecu-lar architecture (comprising just one polypeptide chain) and they do not require post-translational modification In addition, anticalins exhibit robust biophysical properties and can easily be produced in microbial expression systems As their structure–function relationships are well understood, rational engineering of additional features such as site-directed pegylation or fusion with functional effector domains, dimerization mod-ules or even with another anticalin, can be readily achieved Thus, antica-lins offer many applications, not only as reagents for biochemical research but also as a new class of potential drugs for medical therapy

Abbreviations

ApoD, apolipoprotein D; BBP, bilin-binding protein; CDR, complementarity-determining region; CTLA-4, cytotoxic T-lymphocyte antigen-4; NGAL, neutrophil gelatinase-associated lipocalin; Tlc, tear lipocalin; VEGF, vascular endothelial growth factor.

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The lipocalins share a structurally conserved b-barrel

as their central folding motif, which is composed of

eight antiparallel b-strands that wind around a central

axis (Fig 1) At its open end the cup-like structure

supports four loops, which form the entrance to the

ligand pocket The opposite end of the b-barrel is

closed by short loops, and densely packed amino acid

side chains form the hydrophobic core in this region

As another typical feature, a C-terminal a-helix packs

against the b-barrel from one side Despite extremely

low mutual sequence homology, the b-barrel is

struc-turally highly conserved among the lipocalins In

con-trast, the loop region around the ligand-binding site

exhibits large mutual differences, both in amino acid

sequence and length, and in the conformation of the

four polypeptide segments [5]

This structural property reflects the many ligand

specificities observed for this protein family and

resem-bles the hypervariable region that forms the

antigen-binding site of antibodies [6] In the immunoglobulins,

six hypervariable loops, also called

complementarity-determining regions (CDRs), are supported by the

structurally rigid b-sandwich framework of the paired variable domains of the light and heavy chains These CDRs come together at the tips of the Y-shaped mole-cule to form a contiguous interface for antigen bind-ing On the basis of this structural resemblance, lipocalins should offer the same potential for molecu-lar recognition as do antibodies In contrast, natural li-pocalins cannot benefit from the mechanisms of somatic gene recombination and hypermutation, which lead to the vast number of different antibodies gener-ated by the immune system However, the methods of combinatorial biochemistry can be employed in order

to engineer artificial lipocalins with novel specificities for prescribed targets, which were hence dubbed

‘anticalins’ [7,8]

Properties and potential of anticalins

Engineered lipocalins offer several advantages over immunoglobulins Their size, of < 20 kDa, is much smaller than that of antibodies, whose extended molec-ular dimensions hamper efficient tissue penetration

Superposition of Lipocalins

Fig 1 Molecular architecture of human lipocalins and structural variability of their binding sites Ribbon representation of the crystal struc-tures of four human lipocalins: retinol-binding protein (RBP; PDB entry 1RBP), apolipoprotein D (ApoD; PDB entry 2HZQ), tear lipocalin (Tlc; PDB entry 1XKI) and neutrophil gelatinase-associated lipocalin (NGAL; PDB entry 1L6M) Lipocalins share a conserved b-barrel of eight antiparallel b-strands (cyan) The four exposed loops at its open end (red), which form the natural ligand-binding site, exhibit high structural variability, which is illustrated by the superposition shown to the right.

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Furthermore, lipocalins have a rather simple

composi-tion, which is based on a single polypeptide chain In

contrast, antibodies comprise two different

polypep-tides (i.e the light and heavy chains), which leads to

unstable domain association when dealing with small

Fv fragments and which also requires complicated

cloning steps for recombinant expression With four

structurally variable loops, the binding site of

lipoca-lins is less complex and easier to manipulate [5] than

the CDR of antibodies, which is composed of

alto-gether six non-sequential loop segments from both

immunoglobulin chains [6]

Naturally, lipocalins lack the constant Fc region,

which mediates immunological effector functions but

often causes undesired interactions of antibodies while

being crucial only for a few biopharmaceutical

applica-tions Finally, many lipocalins lack glycosylation and

can thus be produced as authentic proteins in

micro-bial expression systems, whereas the manufacture of

glycosylated full-size antibodies requires expensive

eukaryotic cell culture, whose optimization and

fer-mentation is time-consuming and prone to limited

capacities [9] While some of these benefits have also

been claimed for engineered single-chain variable

frag-ments of antibodies or isolated VHH domains of

cam-eloid immunoglobulins, for example their practical

applicability compared with intact antibodies,

espe-cially for medical purposes, is still unclear [10]

Similarly to the immunoglobulins, human lipocalins

occur as soluble proteins in the plasma and other

tissue fluids, with concentrations up to approximately

1.0 mgÆmL)1 Most of the lipocalins are freely

distrib-uted in the body, where they exert a ligand buffer or

transport function This predestines this family of

proteins not only as carrier vehicles or scavengers for

pharmaceutically active compounds but also, especially

when engineered for novel binding functions, as

thera-peutic drugs on their own [11]

Both natural and engineered lipocalins are often

surprisingly stable, with melting temperatures above

70C [12], and they are easily produced in Escherichia

coli in a functional state [4] The recombinant

lipoca-lins can be recovered as soluble monomeric proteins,

even when lacking natural glycosylation (eg: ApoD

and NGAL) Lipocalins are typical secretory proteins,

both in vertebrates and in lower organisms such as

insects, and thus they often carry one or two

disul-phide bonds Consequently, bacterial production via a

secretory route is the method of choice [4,13], albeit

several recombinant lipocalins were also successfully

isolated from the soluble cytoplasmic extract of E coli

[14,15] As the disulphide bridges are not buried in the

hydrophobic interior of lipocalins – but rather serve

for cross-linking the N- and C-terminus to the b-barrel [5] – they are not as crucial for folding as is the case for immunoglobulins Indeed, several natural lipocalins devoid of disulphide bonds exist (eg: the human epi-didymal and the bacterial lipocalins), and in other li-pocalins (e.g Tlc) the single disulphide bond can be eliminated without much loss of protein stability Interestingly, especially among the human lipocalins, many members carry an additional free Cys residue Its reactive thiol side chain sometimes serves for cross-linking to other plasma proteins, although the physiological function is often not known Thus, for application as research reagents, or in medical diagnos-tics as well as therapy, it is usually advisable to substi-tute the unpaired Cys residue with an inert amino acid, such as Ser [4] On the other hand, a free Cys res-idue can be used for the site-specific covalent attach-ment of functional groups via maleimide chemistry, including fluorescent labels or poly(ethylene glycol), which can serve for plasma half-life extension [16] Lipocalins are also well suited for the construction of functional fusion proteins The fusion of anticalins with alkaline phosphatase, for example, leads to useful reporter reagents [17] Anticalins may even be fused with each other, yielding either bivalent or bispecific-binding proteins, so-called ‘duocalins’ [18]

Anticalins recognizing small molecules

Initially, the structurally and biochemically well char-acterized bilin-binding protein (BBP) of Pieris brassi-cae[19] was employed to engineer an artificial binding site for ligands such as fluorescein and digoxigenin, as well as other small molecules and peptides This lipoc-alin comprises 174 residues and exhibits a rather wide and shallow ligand pocket, where biliverdin IXc is complexed as natural ligand Sixteen residues distrib-uted across all four loop segments, whose side chains form the centre of the binding site, were identified by molecular modelling and subjected to concerted random mutagenesis, followed by phagemid display selection for variants with novel binding activities [7]

In the case of fluorescein, which was chosen as a well-known immunological hapten, several variants with high specificity and dissociation constants as low

as 35.2 nm were identified Following X-ray structural analysis of the complex between the engineered lipoca-lin and this ligand [20], improved variants with KD

values for fluorescein of approximately 1 nm were rationally engineered just by optimizing two side chains in the binding pocket [21] Thus, it was demon-strated that engineered lipocalins with novel specifici-ties (i.e anticalins) can provide hapten-binding

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proteins with affinities in a range that was so far

con-sidered typical for antibodies Notably, the BBP

vari-ants appeared to recognize fluorescein – or other small

molecule targets – as true haptens, without measurable

context-dependence concerning the carrier protein that

served for ligand immobilization during the

phage-dis-play panning process With their ability to provide

deep and highly complementary ligand pockets,

antica-lins distinguish themselves from most other protein

scaffolds that are currently under investigation [22]

From the same BBP mutant library an anticalin with

specificity for the cardiac steroid digoxigenin was

selected [17] Its initially moderate affinity was

subse-quently raised by selective random mutagenesis of the

first hypervariable loop, followed by phagemid display

and colony screening under more stringent conditions,

thus resulting in a 10-fold improved KD value of

30.2 nm Attempts to raise the affinity for digoxigenin

even further were made in a combinatorial approach

using a ‘loop-walking’ randomization strategy [12] and

also by rational protein design based on the crystal

structure of this engineered lipocalin [23] These

approaches allowed the identification of several point

mutations, leading to KD values as low as 800 pm for

digoxin (i.e the natural glycosylated derivative of

digoxigenin) [11]

The crystal structures of these first anticalins in

com-plex with their ligands – and in one instance also as

apo-protein – which were solved at resolutions of

2.0 A˚ or better [20,23], provided interesting insight into

the mechanism and specificity of molecular recognition

by engineered lipocalins (Fig 2) Most importantly, the extensive replacement of side chains, affecting 10%

of all residues in the BBP, did not impair the b-barrel fold The randomized loops, on the other hand, adopted dramatically altered conformations compared with the wild-type lipocalin Both fluorescein and digoxigenin are bound at the bottom of the cleft that harbours biliverdin IXc in the BBP [24] Thus, while the overall topology of the lipocalin, comprising the b-barrel with the a-helix attached to it, remained con-served for both anticalins, the set of four loops at the entrance to the ligand pocket exhibited pronounced conformational differences in comparison with each other and with the BBP These structural changes seem

to be triggered by the amino acid substitutions that were introduced during the combinatorial engineering

of the anticalins rather than by complex formation with the ligand, thus illustrating the inherent structural plasticity of the lipocalin loop region

Indeed, the mechanism of complex formation, at least with low-molecular-weight ligands, appears to be similar to the interaction between antibodies and hap-tens, except that the ligand can be buried more deeply

in the engineered lipocalin pocket Shape complemen-tarity is mainly generated by means of aromatic side chains, and specific interactions arise from suitably placed hydrogen-bond donors or acceptors, sometimes mediated by buried water molecules Notably, in the case of the digoxigenin-binding anticalin, DigA16, the bound steroid ligand is sandwiched between one Trp and two Tyr side chains, very similar to a monoclonal antibody directed against digoxin, which provides a nice example of ‘convergent’ in vitro evolution [23] In addition, a His side chain at the bottom of the ligand pocket displays an induced fit upon complex formation with digoxigenin, an effect so far regarded as common

in antibodies Further to the pronounced backbone plasticity in the loop region, comparison of the pri-mary sequences of many engineered lipocalins revealed that all randomized amino acid positions essentially tolerate the entire set of natural side chains

Apart from these fundamental insights into the struc-ture–function relationships of lipocalins and their simi-larity to immunoglobulins, the resulting anticalin, which was designated Digical, may be applicable as a therapeutic agent for the treatment of digitalis intoxica-tions Although digitalis is widely applied in conjunc-tion with heart insufficiency and arrhythmias [25], it has a very narrow therapeutic window, and precise adjustment of digoxin plasma levels is mandatory to prevent poisoning with fatal outcome Indeed, when Digical was employed for studies in a guinea-pig animal model of digitalis intoxication, the anticalin appeared

NLoop #4

Loop #1

C Loop #2

Loop #3

Fig 2 3D structure of an anticalin in complex with its cognate

ligand Ribbon representation of the crystal structure of the

digoxi-genin-binding anticalin DigA16 (PDB entry 1LKE) The bound ligand

is shown in a space-filling representation in yellow, whereas the 16

amino acid side chains in the four hypervariable loops – as well as

the adjoining regions of the b-barrel – which were randomized in

the naive combinatorial library derived from the BBP used for the

anticalin selection, are depicted in orange The N-terminus (N) and

the C-terminus (C) of the polypeptide chain are labelled.

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to be effective in reversing the digoxin-induced toxicity

after administering just a moderate stoichiometric

excess [11], thus demonstrating the acute protective

effect of this anticalin on the cardiovascular system and

its suitability as an antidote against digoxin

Furthermore, the anticalin FluA, which possesses

high affinity for fluorescein, has the interesting

prop-erty of almost completely quenching the fluorescence

emission of this widely applied reagent [7] The reason

for the disappearance of the stationary ligand

fluores-cence seems to be an ultrafast electron transfer

between the excited fluorescein dianion and a Trp side

chain in the binding site of the engineered lipocalin,

which closely packs against the xanthenolone moiety

[26] This phenomenon opens interesting applications

in biophysics Such an ‘anti-fluorescent’ protein could

also be useful as a reagent for the specific quenching

of background signals that arise from fluorescein

groups surrounding a cell, for example when

deter-mining the topology of a site-specifically labelled

membrane protein

Anticalins directed at proteins

Considering medical applications, extracellular proteins

or cell-surface receptors are the predominant class of

biomolecules that currently provide relevant targets for

biopharmaceuticals such as antibodies Consequently,

in recent years anticalin libraries were specifically

developed for the recognition of such protein

‘anti-gens’ In addition, to reduce immunogenic side effects

upon prolonged treatment, these libraries were

con-structed on the basis of natural human lipocalins, in

particular ApoD [27,28], NGAL [29] and Tlc [30]

(Fig 1)

To this end, 16–24 amino acid residues located at

exposed positions, close to the tips of the four

hyper-variable loops, were subjected to random mutagenesis

in order to allow tight contact formation with a

mac-romolecular target, which cannot penetrate as deeply

into the ligand-binding site as a small molecule Using

these libraries, anticalins with high specificity and

affinities in the subnanomolar range were successfully

selected against a variety of disease-related protein

antigens, including immunological receptors such as

cytotoxic T-lymphocyte antigen-4 (CTLA-4) [11] and

soluble growth factors such as vascular endothelial

growth factor (VEGF) [31]

Recently, the crystal structure of the complex

between a cognate anticalin and the extracellular

domain of CTLA-4 was solved, demonstrating that a

macromolecular ‘protein antigen’ can be effectively

bound at the cup-shaped binding site of an engineered

lipocalin, even though its natural counterparts almost exclusively recognize low-molecular-weight substances All four randomized loops of NGAL – which had served as a lipocalin scaffold in this case – contribute

to the formation of the molecular complex, thus vali-dating the design of the anticalin library

CTLA-4 (CD152) is an activation-induced, trans-membrane T-cell coreceptor with an inhibitory effect

on T-cell-mediated immune responses [32] CTLA-4 antagonizes the CD28-dependent costimulation of T cells, whereby CTLA-4 and CD28 share the same counter-receptors on antigen-presenting cells (i.e B7.1 and B7.2) Notably, the bound anticalin shields the CTLA-4 epitope that is involved in the interaction both with B7.1 and B7.2 Indeed, an antagonistic activity of the anticalin towards CTLA-4 was con-firmed in several in vitro cell culture tests, where T-cell proliferation was stimulated in a manner comparable

to that of commercially available antibodies directed against the same target Thus, the CTLA-4-specific anticalin is a promising drug candidate for the immu-notherapy of cancer, similarly to corresponding anti-bodies that are already in clinical trials [33] Apart from its much smaller size and probably better tissue penetration, the lack of immunological effector func-tions – which reside in the antibody Fc region – for the anticalin should limit off-target toxicity because only the antagonistic activity is needed In fact, this is the case for many relevant targets involved in the regu-lation of the immune response and inflammation as well as neoangiogenesis

Another promising drug candidate is an anticalin with strong antagonistic activity towards VEGF VEGF is a well-characterized mediator of tumor angiogenesis and other neovascular diseases [34], for example age-related macular degeneration (AMD) The selected anticalin exhibits a favorable binding and activity profile in direct comparison with currently approved VEGF antagonists [31] A half-life extended version of the anticalin has demonstrated excellent effi-cacy in three animal models assessing VEGF-induced enhanced vascular permeability, angiogenesis and anti-xenograft tumor activity As immunological effector functions again appear to be irrelevant for biomedical activity, an anticalin with proven VEGF-antagonistic function should offer an interesting alternative to full-size antibodies, especially in the light of its presumably better distribution

Conclusions and prospects

Engineered lipocalins offer binding sites with surpris-ingly high structural plasticity and an extended

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molecular interface for target recognition which is

comparable in size to that of antibodies Anticalins

with high specificity and affinity, down to picomolar

dissociation constants, can be readily generated against

haptens, peptides and proteins Thus, regarding their

range of addressable targets they surpass other protein

scaffolds that are presently pursued [22] Available

structural and functional data suggest that anticalins

are able to recognize a diverse set of epitopes on

differ-ent target proteins and therefore have considerable

potential as specific antagonistic reagents in general

Consequently, anticalins constitute promising

reagents for therapeutic applications As anticalins

can be derived from human lipocalin scaffolds, the

risk of immunogenicity is minimized and further

reformatting – such as by CDR grafting for the

‘humanization’ of antibodies – is not required The

absence of immunological effector functions prevents

many potential side effects known for antibodies

Furthermore, the monovalent binding activity of

anticalins decreases the risk of intermolecular

cross-linking of cellular receptor targets that could lead to

unwanted signal triggering

Natural lipocalins, as well as engineered lipocalins,

are quickly cleared by renal filtration, as a result of

their small size of approximately 20 kDa, if they

circu-late as monomeric proteins When conjugated with

radioactive isotopes for in vivo diagnostics, for

exam-ple, such properties should lead to images of high

contrast soon after administration Nevertheless, for

medical indications that require prolonged treatment,

the simple architecture and robustness of the lipocalin

scaffold facilitates the preparation of fusion proteins

or of site-directed conjugates to decelerate clearance

In principle, several established techniques are

avail-able to extend the plasma half life of anticalins, for

example by the production of fusion proteins with

serum albumin, with an albumin-binding domain or

peptide or via pegylation

Anticalins display both their N-terminus and

C-ter-minus in an accessible manner and remote from the

binding site, which differs from the situation with

sin-gle-chain variable fragments of antibodies, where the

N-terminus often forms part of the paratope Thus,

anticalins are well suited for fusion with other

func-tional domains without compromising their engineered

binding activities Fusion proteins of anticalins that

address a specific receptor on solid tumors with

enzymes which generate a cytotoxic compound from

an inactive precursor (prodrug) might be of special

interest as an alternative to antibody-directed enzyme

prodrug therapy (ADEPT) Furthermore, a dimeric

binding mode utilizing either a duocalin that has twice

the same target specificity or a fusion protein between

an anticalin and a dimerization domain may be employed to enhance binding avidity

Hence, owing to their adaptable binding site and their simple and robust molecular architecture, specifi-cally engineered anticalins promise a future as versatile reagents for research, biotechnology and medicine

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