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Based on a sequence identity matrix of the deduced amino acid sequences, we divided the obtained sequences into five groups, naming them macrocypin 1–5.. Sequence identity at the deduced

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the basidiomycete Macrolepiota procera

Jerica Saboticˇ1, Tatjana Popovicˇ1, Vida Puizdar2and Jozˇe Brzin1

1 Department of Biotechnology, Jozˇef Stefan Institute, Jamova 39, Ljubljana, Slovenia

2 Department of Biochemistry and Molecular and Structural Biology, Jozˇef Stefan Institute, Jamova 39, Ljubljana, Slovenia

Introduction

Papain-like cysteine proteases are widespread in

organ-isms ranging from bacteria to humans They play

impor-tant roles in many facets of physiology, and

dysregulation of proteolytic activity can lead to a variety

of pathologies, including cancer, rheumatoid arthritis,

osteoarthritis and neurological disorders [1,2] The most

important regulators of protease activity are specific

protease inhibitors In addition to their considerable

potential in diverse medical applications, protease

inhib-itors have been studied as tools for analysing proteolytic mechanisms and protein–protein interactions, and as biocidal agents against various organisms There are several groups of inhibitors, mainly from animal and plant origins, that specifically inhibit papain-like cyste-ine proteases [3,4] The first cystecyste-ine protease inhibitor isolated from higher fungi was clitocypin from the basidiomycete Clitocybe nebularis [5] Clitocypin is a 16.8-kDa protein lacking cysteine and methionine

resi-Keywords

basidiomycetes; clitocypin; cysteine

protease; mycocypin; protease inhibitor

Correspondence

J Saboticˇ, Department of Biotechnology,

Jozˇef Stefan Institute, Jamova 39, 1000

Ljubljana, Slovenia

Tel: +386 1 477 3754

Fax: +386 1 477 3594

E-mail: Jerica.Sabotic@ijs.si

Note

The nucleotide sequences reported in this

paper have been deposited in the

DDBI ⁄ EMBL ⁄ GenBank databases under

accession numbers FJ495239, FJ495240,

FJ495241, FJ495242, FJ495243, FJ495244,

FJ495245, FJ495246, FJ495247, FJ495248,

FJ495249, FJ495250, FJ548751 and

FJ548752

(Received 25 March 2009, revised 22 May

2009, accepted 9 June 2009)

doi:10.1111/j.1742-4658.2009.07138.x

A new family of cysteine protease inhibitors from the basidiomycete Mac-rolepiota procera has been identified and the family members have been termed macrocypins These macrocypins are encoded by a family of genes that is divided into five groups with more than 90% within-group sequence identity and 75–86% between-group sequence identity Several differences

in the promoter and noncoding sequences suggest regulation of macrocypin expression at different levels High yields of three different recombinant macrocypins were produced by bacterial expression The sequence diversity was shown to affect the inhibitory activity of macrocypins, the heterolo-gously expressed macrocypins belonging to different groups showing differ-ences in their inhibitory profiles Macrocypins are effective inhibitors of papain and cysteine cathepsin endopeptidases, and also inhibit cathepsins

B and H, which exhibit both exopeptidase and endopeptidase activities The cysteine protease legumain is inhibited by macrocypins with the excep-tion of one representative that exhibits, instead, a weak inhibiexcep-tion of serine protease trypsin Macrocypins exhibit similar basic biochemical characteris-tics, stability against high temperature and extremes of pH, and inhibitory profiles similar to those of clitocypin from Clitocybe nebularis, the sole rep-resentative of the I48 protease inhibitor family in the MEROPS database This suggests that they belong to the same merops family of cysteine prote-ase inhibitors, the mycocypins, and substantiates the establishment of the I48 protease inhibitor family

Abbreviations

AMC, 7-amido-4-methylcoumarin; Clt, clitocypin; Mcp1, 2, 3, 4, 5, macrocypin 1, 2, 3, 4, 5; rMcp1, 3, 4, recombinant macrocypin 1, 3, 4; Z, benzyloxycarbonyl.

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dues, which, on account of its unique characteristics,

was assigned to a new family of cysteine protease

inhibi-tors, I48 in the merops database inhibitor classification,

also named mycocypins Its profile of inhibition differs

from those of other then known families of cysteine

pro-tease inhibitors Clitocypin inhibits papain, cathepsins L

and K, legumain and bromelain, but is inactive against

cathepsin H, trypsin and pepsin Clitocypin is encoded

by a small family of genes that show sequence

hetero-geneity, which does not affect its inhibitory activity

In addition to a defensive role, a regulatory role in

mushroom endogenous proteolytic systems was

pro-posed, based on the specific inhibition of several

puta-tive fungal cysteine proteases [5–7]

The proteolytic potential of higher fungi is

consid-erable in terms of the number and diversity of

prote-ases they contain [8], and basidiomycetes are a rich

source of novel proteolytic enzymes and their

inhibi-tors [5,8] As a result of a study carried out to

inves-tigate the extent and function of the I48 inhibitor

family that to date includes only one characterized

member, we report a novel family of cysteine

prote-ase inhibitors from basidiocarps, or fruiting bodies,

of the basidiomycete Macrolepiota procera, and have

characterized some of its natural and recombinant

members

Results

Isolation of cysteine-protease inhibitors from

M procera

Cysteine protease inhibitors were purified from

basid-iocarps of M procera by a method similar to that used

to purify clitocypin from basidiocarps of C nebularis,

which included hydrophobic interaction, ion-exchange

and papain affinity chromatographies [5,9] The

inhibi-tory proteins were separated on SDS–PAGE under

denaturing, nonreducing, conditions into two bands

corresponding to apparent molecular masses of 21 and

17 kDa (Fig 1) The 43-residue N-terminal sequence

for the former protein (Fig 2) was 42% identical and

58% similar to the N-terminal sequence of clitocypin

The new 21-kDa cysteine protease inhibitor has been

named macrocypin (Macrolepiota procera cysteine

pro-tease inhibitor, Mcp) The N-terminal sequence for the

lower-molecular-mass protein could not be determined,

possibly because of a blocked N terminus The

pres-ence of a protein of similar size to that of clitocypin

and purified by the same procedure, and with similar

biochemical properties, indicated the presence of a

clit-ocypin-like cysteine protease inhibitor in M procera

[5,6] This was confirmed at the genetic level by

geno-mic DNA dot-blot analysis and by amplification of partial clitocypin-like gene sequences The partial sequences of the clitocypin-like genes from M procera are more than 90% identical to those of clitocypin

at the nucleotide level (see Supplementary Data in Doc S1 and Doc S2 and Fig S1 and S2)

Cloning and analysis of macrocypin cDNA and gene sequences

The N-terminal sequence (H2N-GLEDGLYTIRHLVE

the sequence of an internal peptide fragment obtained

by digestion with cyanogen bromide (H2 N-YIP-RKVFK) were used to design degenerate primers (Table S1) Degenerate primers and M procera cDNA synthesized from total RNA as template were used to obtain a specific macrocypin sequence This was then used to design specific nested primers for use in Genome walking and 3¢ RACE methods

Two fragments were cloned from the two genomic libraries obtained by the Universal Genome Walker kit (Clontech, Heidelberg, Germany), using mcp gene-spe-cific antisense primers – Pr4 (1000 bp) and Pr3 (204 bp) Each corresponds to the mcp 5¢ UTR and promoter regions Sequences spanning the 5¢ coding region of the mcp gene were not identical (Fig S3), suggesting that more than one gene encoding macrocy-pin is present in the M procera genome, each of which has its corresponding promoter Both promoter sequences have a typical TATA box (TATAAAA) present at position )85, and a putative transcription initiation site (CTAGTCC) at position )55, indicating

Fig 1 SDS–PAGE analysis of the natural cysteine protease inhibi-tors clitocypin (nClt) and macrocypin (nMcp) Clitocypin (from Clito-cybe nebularis) and macrocypin (from Macrolepiota procera) purified from basidiocarps were analyzed under nonreducing, dena-turing conditions and stained with Coomassie Blue Lane M, protein molecular mass markers.

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transcriptional activity of macrocypin genes The

loca-tion of the TATA box at a posiloca-tion 30 nucleotides

upstream from the putative transcription-initiation site

is in accordance with the locations found in several

other fungal genes, which are usually within 30–60

nucleotides of the transcription start site [10]

To obtain the coding and 3¢ UTR regions of the mcp

mRNA, we used the 3¢ RACE method with mcp-specific

sense primers that differentiate between the two

sequences obtained by genome walking Partial

sequences corresponding to promoter Pr4 showed two

different lengths of the 3¢ UTR (66 nucleotides and 87

nucleotides) Sequences corresponding to promoter Pr3

showed even more variation in 3¢UTR length (64, 69, or

88 nucleotides), indicating the possibility of macrocypin

translation being regulated via the 3¢ UTR [11] There is

no typical polyadenylation signal present in the 3¢ UTR

The genomic sequence corresponding to the 3¢ UTR was

amplified from the two genomic libraries created using

the Universal Genome Walker kit (Clontech), using

mcp-specific sense primers Two fragments, spanning

175 and 151 bp downstream of the stop codon, were

obtained that were identical to the overlapping sequence

of the 3¢UTR region amplified from M procera cDNA,

corresponding to the promoter fragment Pr4

The full-length gene and cDNA sequences of macro-cypin were obtained, using primers annealing to the 5¢ UTR and 3¢ UTR regions and genomic DNA or first-strand cDNA synthesized from the total RNA of

M procera as templates Several different gene and cDNA sequences were amplified, all of which, how-ever, shared the same gene structure The mcp genes were found to be composed of four exons and three short introns with exon–intron boundaries matching the consensus splice sites predicted for eukaryotic genes [10] Based on a sequence identity matrix of the deduced amino acid sequences, we divided the obtained sequences into five groups, naming them macrocypin 1–5 Sequence identity at the deduced amino acid sequence level between the five macrocypin groups was 75–86%, while sequences within groups exhibited more than 90% sequence identity

The length of the macrocypin 1 (Mcp1) deduced amino acid sequence was 169 residues, with a molecu-lar mass of 19 193 Da, while the length of the repre-sentatives of the other four groups was 167 residues, with molecular masses between 18 770 and 19 031 Da Single cysteines were present in Mcp1, macrocypin 4 (Mcp4) and macrocypin 5 (Mcp5) (C106), none in macrocypin 2 (Mcp2) and none or one (C75) in

macro-Fig 2 Diversity in the cysteine protease inhibitor macrocypin family Amino acid sequences deduced from macrocypin (Mcp) genes (pref-aced by g) and from cDNA sequences (pref(pref-aced by c) belonging to different groups (macrocypins 1–5) are aligned with the N-terminal sequence determined for the natural macrocypin (nMcp) isolated from basidiocarps of Macrolepiota procera Identical residues in at least 9

of 11 sequences (80 %) are highlighted in dark gray and similar residues are highlighted in light gray Sequences corresponding to clones used for the heterologous expression of macrocypins are indicated in bold Residues subjected to positive evolution, as determined using the Datamonkey rapid detection of positive selection web server [20], are marked with an asterisk.

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cypin 3 (Mcp3) All the sequences showed high

con-tents of proline, of around 9.2% (the average overall

protein content is 4.7 %), of tryptophan, of 3.6%

(average 1.1 %) and of tyrosine, of 6.0–6.6% (average

2.9%), and low leucine contents of around 3.6%

(aver-age 9.7%) [12] With the exception of Mcp3,

macrocy-pin sequences also showed a high glycine content of

around 9.8% (average 6.9%)

Variability in the sequences of macrocypin genes

The diversity observed for the macrocypin coding

sequences was greater than that for clitocypins [7] The

coding sequence, composed of four exons, was 507 bp

for Mcp1 and 501 bp for the other macrocypins The

deduced amino acid sequence for Mcp1 was thus two

amino acids longer, because of an insertion near the N

terminus The lengths of the four exons were 147 or

153, 212, 70 and 75 bp, and of the three introns were

56, 49 or 51, and 50 or 54 bp The sequence diversity

was equally distributed in introns and in exons

(Fig S3) The second intron showed the greatest

diver-sity in sequence and was 2 bp shorter in Mcp3 and

Mcp5 The third intron was the most highly conserved

of the three, with the exception of Mcp3, where it was

4 bp shorter Diversity in the coding sequence was

dis-tributed throughout the sequence (Fig 2) and this was

caused by one, two or three nucleotide substitutions

that resulted in 47 variable codons at the level of the

deduced amino acid sequences Some of these codons

subjected to positive selection (codons 18, 85, 89, 105,

107 and 116) were identified at the P < 0.05 level (Fig 2) Codons under negative selection (31 codons identified) appeared to be evenly distributed along the whole mcp genes without particular clustering The few positively selected sites in the mcp genes may provide insights into the physiological role of macrocypins, as these sites are more likely to be involved in interac-tions with other proteins

Similarity searches for the macrocypin sequences against NCBI databases using blastn [13] revealed no significant similarities (cut-off e < 0.1) tblastn searches, by contrast, found significant sequence simi-larity to the clitocypin cysteine protease inhibitor annotated in the Laccaria bicolor S238N-H82 genome [14] There was 39–44% sequence similarity and 26–29% sequence identity for different macrocypin sequences, the highest being with that of Mcp5 No significant similarities were found for the macrocypin sequences in the completed and unfinished archaeal or bacterial genomes or in other eukaryotic genomes, which is probably the result of low overall sequence similarity between mycocypins

Alignment of macrocypin deduced amino acid sequences with clitocypin sequences showed 17–21% sequence identity (Fig 3) The N-terminal halves of the sequences showed more similarity The higher molecular masses relative to the clitocypins are caused

by a few insertions and deletions of two to seven amino acids, distributed along the sequence Another important difference between the macrocypin and clit-ocypin sequences was the presence of a cysteine residue

Fig 3 Alignment of mycocypin deduced amino acid sequences Deduced amino acid sequences of macrocypins belonging to each of the five groups are aligned with three representative clitocypin deduced amino acid sequences (GenBank accession numbers: gClt-Kras, AAZ78483.1; cClt-Kras, AAZ78481.1; and cClt-Vrh, AAZ78482.1) Sequences are prefaced by g or c to indicate whether they are deduced from genomic or cDNA sequences Identical residues in at least seven of the eight sequences (90 %) are highlighted in dark grey and similar residues are highlighted in light grey.

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in most macrocypins, and of several histidine and

methionine residues in all the macrocypin sequences,

all of which are absent in clitocypin High proline and

glycine contents were, by contrast, common to both

macrocypins and clitocypin

Heterologous expression of macrocypins

The cDNA clones that were available for Mcp1, Mcp3

and Mcp4 (marked in bold in Fig 2), were cloned into

expression vectors of the pET System (Novagen,

Madi-son, WI, USA) for heterologous expression in

Escheri-chia coli (Fig S4) Macrocypin 1a (Mcp1a) and

macrocypin 3a (Mcp3a) cDNA clones were each

cloned into two expression vectors (pET3a and

pET11a) to assess their expression in the bacterial

expression system Heterologous expression of

recom-binant Mcp1 (rMcp1) was higher with the pET11a

expression vector construct, while that of recombinant

Mcp3 (rMcp3) was higher using pET3a The amounts

of both recombinant proteins were highest 6 h after

induction (data not shown) The macrocypin 4a

(Mcp4a) cDNA clone was introduced into the pET14b

expression vector and expressed in two strains of

E coli The expression of recombinant Mcp4 (rMcp4)

was highest when using the pET14b::Mcp4a construct

in combination with the E coli BL21(DE3) pLysS

strain grown for 8 h after induction Recombinant

macrocypins rMcp1 and rMcp3 were expressed mainly

as insoluble inclusion bodies, and rMcp4 was

expressed as an equal distribution of protein between

the soluble form and inclusion bodies All three rMcps

were purified from the inclusion bodies, which were

almost completely solubilized in 3 m urea One-step

purification using size-exclusion chromatography

yielded purified recombinant macrocypins rMcp1 and

rMcp4, while rMcp3 still contained some impurities,

which were taken into consideration when calculating

the concentration

Characterization of macrocypins

Recombinant macrocypins rMcp1, rMcp3 and rMcp4

were resolved on SDS–PAGE under reducing

condi-tions as single 19 kDa bands (Fig 4A) Under

non-reducing conditions, however, they all showed an

additional band at 38 kDa, corresponding to a

dimer, probably formed between the single cysteines

present in each protein The calculated isoelectric

point of 5.1 for Mcp4 was confirmed by IEF and

was similar to the isoelectric point of the natural

macrocypin isolated from basidiocarps of M procera

For Mcp1 and Mcp3, the calculated isoelectric points

were 4.8, which was also confirmed by IEF (Fig 4B)

N-terminal sequences were confirmed for all three recombinant macrocypins That for rMcp1 [NH2 -(M)GFEDG] revealed N-terminal cleavage of methio-nine in approximately one-third of the molecules N-terminal sequences of rMcp3 and rMcp4 (NH2 -ALEDG) showed complete cleavage of N-terminal methionine N-terminal cleavage of methionine in

E coli is strongly influenced by the amino acid resi-dues at the P1¢, P2¢ and P3¢ positions (the P1 position being the first methionine) [15] In the case of macro-cypins, the NH2-MALE sequence favours N-terminal methionine cleavage in E coli, and the NH2-MGFE sequence of rMcp1 favours only partial cleavage The far-UV CD spectra of rMcp1 and rMcp4 con-firmed the expectation from the sequences that the conformations of the inhibitors are very similar (Fig S5A) The marked tryptophan bands, seen also

in clitocypin [9] and ascribed to interaction of buried tryptophan residues, prevent analysis of secondary structure, but underline the similarity in tertiary struc-ture, at least in this region, as well as of secondary structure

Clitocypin has been proven to be a very stable pro-tein [5,9] The temperature and pH stability of recom-binant macrocypins were determined by following their inhibitory activity (measured after return to native conditions) after heating and after incubation at extremes of pH The macrocypins rMcp1 and rMcp3 retained their inhibitory activity after heating at 75 C,

or even at 100C, for 15 min, whereas rMcp4 partially lost its inhibitory activity after heating at 75 C for

15 min and completely lost its inhibitory activity after

Fig 4 Comparison of natural and recombinant macrocypins by SDS–PAGE (A) and IEF (B) Purified natural (nMcp) and recombinant macrocypins (rMcp) were subjected to SDS–PAGE analysis under reducing denaturing conditions and to IEF Lane M, protein mole-cular mass markers; lane S, standard protein IEF markers; lane 1, natural macrocypin (nMcp); lane 2, recombinant macrocypin 4 (rMcp4); lane 3, recombinant macrocypin 1 (rMcp1); lane 4, recom-binant macrocypin 3 (rMcp3).

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heating at 100C Similarly, rMcp1 and rMcp3

retained their inhibitory activities after incubation in

acidic (pH 2) or alkaline (pH 11) conditions While

rMcp4 lost its inhibitory activity after incubation at

pH 2, incubation at pH 11 had no influence

Stability was also examined by following conforma-tion directly by CD rMcp1 and rMcp4 were unfolded

on thermal denaturation, with closely similar transi-tions, with temperature midpoints of 78C (Fig S5B) The similarity of this value to that for clitocypin shows that the differences in sequence between these three inhibitors are not critical for the stability of the pro-teins Combined, the above results show that the inhib-itors unfold reversibly Measurements of CD spectra at

pH 2.2 and pH 11 showed only a slight decrease in ellipticity after 24 h of exposure

Specificity of macrocypins The inhibitory specificities of the natural macrocypin isolated from basidiocarps and the three recombinant macrocypins were determined against several cysteine proteases Association (ka) and dissociation (kd) rate constants and equilibrium constants (Ki) were deter-mined by continuous assays for papain, cathepsins L and V, and legumain (Table 1), for which typical biphasic progress curves were obtained For inhibition

of cathepsins S, K, B and H and trypsin by macrocy-pins, only equilibrium constants were determined (Table 2) Macrocypins 1 and 3 were found to be effec-tive inhibitors of papain, cathepsin L and cathepsin V, with a mean Ki value of 0.5 nm Macrocypin 4 was also an effective inhibitor of papain but exhibited weaker inhibition of cathepsin L and V than the other two recombinant macrocypins The sample of natural macrocypin showed weaker inhibition for these

Table 1 Inhibition of cysteine proteases by natural and

recombi-nant macrocypins Kinetic and equilibrium constants for the

inhibi-tion of papain, cathepsins L and V, and legumain were determined

under pseudo-first-order conditions in continuous kinetic assays at

25 C and calculated by nonlinear regression analysis according to

Morrison [32] Standard deviation is given where appropriate; ND,

not determined; nMcp, natural macrocypin; rMcp1, recombinant

macrocypin 1; rMcp3, recombinant macrocypin 3; rMcp4,

recombi-nant macrocypin 4.

Enzyme 10)6k a ( M )1Æs)1) 104

k d (s)1) K i (n M ) nMcp

Cathepsin L 0.33 ± 0.11 1.25 ± 0.26 3.81 ± 1.66

Cathepsin V 0.08 ± 0.01 9.85 ± 1.54 12.6 ± 3.8

rMcp1

Cathepsin L 5.52 ± 0.51 35.1 ± 3.2 0.64 ± 0.22

Cathepsin V 1.48 ± 0.01 10.3 ± 0.7 0.69 ± 0.06

rMcp3

Cathepsin L 3.58 ± 0.45 11.1 ± 0.5 0.31 ± 0.06

Cathepsin V 1.88 ± 0.09 8.43 ± 0.76 0.45 ± 0.01

Legumain 0.063 ± 0.020 5.77 ± 1.27 9.17 ± 1.09

rMcp4

Cathepsin L 1.62 ± 0.55 45.1 ± 5.1 2.76 ± 0.92

Cathepsin V 2.29 ± 0.66 33.3 ± 6.3 1.44 ± 0.11

Table 2 Inhibition of various proteases by natural and recombinant mycocypins Equilibrium constants for the inhibition of different prote-ases were determined in continuous or stopped kinetic assays and analyzed according to Morrison [32] or Henderson [33], respectively Kinetic data for the interaction of clitocypin with papain, cathepsins L, K and H, and legumain were reported previously [6] Standard devia-tion is given where appropriate nMcp, natural macrocypin; rMcp1, recombinant macrocypin 1; rMcp3, recombinant macrocypin 3; rMcp4, recombinant macrocypin 4; nClt, natural clitocypin; rClt, recombinant clitocypin; n.i., no inhibition.

Enzyme

Ki(n M )

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enzymes than the recombinant macrocypins Inhibition

of cathepsins S and K by recombinant macrocypins

was somewhat weaker, with Ki values in the range of

5–25 nm, and 10 times higher for inhibition of

cathep-sin K by rMcp1 Cathepcathep-sin B, which has both

endo-peptidase and exoendo-peptidase activities, was inhibited by

macrocypins with Ki values in the micromolar range,

with the exception of rMcp3 (Ki> 1 lm) By contrast,

cathepsin H was inhibited by natural macrocypin and

rMcp1, with Ki values in the micromolar range, while

rMcp3 and rMcp4 inhibited with 10-fold lower values

Like clitocypin, recombinant macrocypins inhibited the

cysteine protease legumain, a member of the C13

fam-ily, with an average Ki value of 6 nm The natural

macrocypin exhibited only very weak inhibition of

legumain (Ki in the micromolar range), while rMcp4

showed no inhibition at all The serine protease trypsin

was not inhibited by natural macrocypin, rMcp1 or

rMcp3, while rMcp4 exhibited weak inhibition with a

Ki value in the micromolar range The aspartic

prote-ase pepsin was not inhibited by natural or recombinant

macrocypins

The ability of natural and recombinant clitocypins

[6] to inhibit cathepsins V, S and B was also

deter-mined for comparison (Table 2) Clitocypin is an

effec-tive inhibitor of cathepsins V and S The weak

inhibition of cathepsin B reported for the natural

clito-cypin, with a Ki value of 0.48 lm [5], was not

con-firmed with either recombinant or natural clitocypin,

for which Kivalues were > 1 lm

Discussion

A novel cysteine protease inhibitor, macrocypin, has

been isolated from basidiocarps of the parasol

mush-room (M procera), in addition to a putative

clito-cypin-like cysteine protease inhibitor Based on

similarities in genetic and biochemical characteristics,

macrocypin was assigned to the mycocypin family of

fungal cysteine protease inhibitors, family I48 in the

merops database, together with clitocypin from

clouded agaric (C nebularis) [6,7]

Based on the partial protein sequence, several

macr-ocypin-coding genes and corresponding cDNAs were

amplified and sequenced, together with the promoter

and 5¢ UTR and 3¢UTR sequences (Fig S3) The

diversity observed was even greater than that observed

for clitocypin-coding genes [7], the macrocypin

sequences being divided into five groups The

variabil-ity was distributed throughout the coding sequence

and comprised one to five consecutive amino acid

sub-stitutions (Fig 2) The variability was not limited to

the coding sequence, as two different promoter

sequences were cloned A few 3¢ UTR sequences were found that showed no sequence diversity, but differed

in their length Different promoter and 5¢UTR and 3¢UTR sequences, and their lengths, together with some differences found in intron sequences and their lengths, which could influence transcription, splicing, mRNA transport, stability and translation, all suggest complex regulation of macrocypin expression at diff-erent levels

Macrocypin- and clitocypin deduced amino acid sequences show similarities, despite a low overall sequence identity of 17–21% (Fig 3) They both have high contents of proline and tyrosine and low contents

of leucine A major difference between clitocypin and macrocypin sequences lies in the presence of sulfur-containing amino acids in macrocypins, but not in clitocypins In spite of very low overall sequence simi-larity, several amino acid residues are conserved in all macrocypin and clitocypin sequences, mainly in the N-terminal half (Fig 3) These are probably important for the inhibitory activity and⁄ or structure, and many

of them are proline residues

A similarity search using macrocypin deduced amino acid sequences against the translated nucleotide data-base at the NCBI server showed significant similarity

to the putative L bicolor clitocypin-like cysteine prote-ase inhibitor Alignment of the deduced sequences of the latter with those of macrocypin from M procera and with clitocypin from C nebularis revealed con-served amino acid residues (Fig S6), the majority of which were already present in the alignment of macro-cypin and clitomacro-cypin deduced amino acid sequences, confirming their functional and⁄ or structural imp-ortance

Macrocypins and clitocypins exhibit similar basic biochemical characteristics They have similar molecu-lar masses of 19 and 16.8 kDa and simimolecu-lar isoelectric points of around 4.8 They both exhibit stability against high temperature and extremes of pH The CD spectrum in the far-UV region for macrocypin, show-ing the characteristic peak around 232 nm that indi-cates an unusually strong contribution of tryptophan residues (Fig S5A), indicates further structural similar-ity to clitocypin [6,9]

The high degree of diversity in macrocypin gene sequences indicates a mixture of inhibitors in the natu-ral macrocypin sample isolated from basidiocarps of

M procera In order therefore to characterize the inhibitors further, recombinant macrocypins were pre-pared Three different macrocypin cDNA clones were used that belong to different macrocypin groups or isoforms Heterologous expression in the bacterial expression system proved successful for all three,

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inhibitory macrocypins (rMcp1, rMcp3, rMcp4) being

obtained by one-step purification from inclusion bodies

with high yields of 50–100 mgÆL)1

The inhibitory profiles of macrocypins and

clito-cypin for several cysteine proteases are similar, but not

identical Of the cysteine proteases tested, macrocypins

inhibit papain and cathepsins L and V most strongly

Compared with clitocypin [6], macrocypins are

stron-ger inhibitors of papain, as a result of higher rate

constants of association, but weaker inhibitors of

cathepsin L, mainly because of increased rate constants

of dissociation Cathepsins S and K are inhibited by

macrocypins with Ki values in the nanomolar range,

while clitocypin inhibits cathepsin K with Kivalues in

the picomolar range The most notable difference

between macrocypin- and clitocypin-inhibition profiles

for cysteine proteases is the weak inhibition by

macro-cypins of cathepsins B and H, the papain-like

prote-ases with endopeptidase and exopeptidase activities As

with clitocypin, legumain, a member of C13 family, is

inhibited by Mcp1 and Mcp3 with Ki values in the

micromolar range The inhibition profile of rMcp4,

with Ki> 1 lm for legumain inhibition and Ki values

in the nanomolar range for papain-like proteases,

con-trasts with those of other macrocypins that inhibit

both papain-like proteases and legumain with Kivalues

in the same range This strongly suggests different

binding sites for the inhibition of the two families of

cysteine proteases Similarly, two independent and

nonoverlapping binding sites have been reported for

family C1 and C13 inhibition by legumain-inhibiting

cystatins C, E⁄ M and F [16]

The ability of macrocypins to inhibit proteases of

other catalytic classes was tested Neither macrocypins

nor clitocypin [5] inhibit the aspartic protease pepsin

The serine protease trypsin is weakly inhibited by

rMcp4, which is the most significant difference in the

inhibitory profiles of macrocypins and clitocypin

The physiological function of the macrocypin

cyste-ine protease inhibitor family is proposed to be defence

against pathogen infection and⁄ or predation by insects

or other pests, analogously to the phytocystatins that

are involved in plant defence by inhibiting exogenous

cysteine proteases during herbivory or infection [17]

The sequence diversity includes amino acid sites of

positive selection The variations in inhibitory profile

between different members of the macrocypin family

reveal different specificities and strengths of inhibition

of cysteine proteases of different evolutionary families,

and even a serine protease These findings together

suggest an adaptation process and the selection of

appropriate inhibitor isoforms providing effective

defence In addition, a regulatory role in intracellular

proteolysis may also be considered for mycocypins, because cysteine protease activity is present in basidio-mycetes and its inhibition by clitocypin was shown in

a few selected basidiomycete species belonging to dif-ferent orders [8]

In conclusion, we have characterized a novel family

of fungal cysteine protease inhibitors – the macrocy-pins – from M procera, at the genetic and biochemical levels and analyzed their inhibition profiles Similarity

to clitocypin from C nebularis is evident at all these levels, suggesting that they belong to the same family

of cysteine protease inhibitors, the mycocypins, thus substantiating the establishment of the I48 family of protease inhibitors in the MEROPS classification that previously comprised only one member In addition to the high conformational stability of mycocypins, their other common characteristic is high genetic diversity, with sequence variability influencing the inhibitory activity in macrocypins, but not in clitocypins Myco-cypins could find use in medical research, as their unique inhibitory profiles could answer the challenge

of finding highly selective inhibitors against proteases important in certain stages of diseases, without affect-ing nontarget proteases Additionally, certain represen-tatives of the macrocypin family, showing inhibition of different classes of proteases, would have applications

in plant protection Double-inhibitory activity against two catalytic classes of proteases in one stable mole-cule could provide more effective protection of plants against insect pests

Experimental procedures

Isolation of cysteine protease inhibitors from

M procera Basidiocarps of the basidiomycete M procera were col-lected from their natural habitat and frozen at )20 C Thawed basidiocarps were homogenized in an equal volume

of 0.1 m Tris–HCl buffer, pH 7.5, containing 0.5 m NaCl, and centrifuged at 8000 g for 30 min Ammonium sulfate was added to the supernatant to a final concentration of 1.3 m before application to a column of Phenyl Sepharose (GE Healthcare, Uppsala, Sweden) After thorough wash-ing with 0.1 m Tris–HCl buffer, pH 7.5, containwash-ing 1.3 m ammonium sulphate, the bound proteins were eluted by a 1.3–0 m gradient of ammonium sulphate in the same buffer Inhibitory fractions, measured against papain, were pooled, concentrated by ultrafiltration (Amicon UM-10; Millipore, Vienna, Austria) and dialyzed against 0.02 m Tris–HCl, pH 7.5 The sample was then applied to a column of DEAE– Sephacel (Pharmacia-LKB, Uppsala, Sweden) equilibrated with 0.02 m Tris–HCl, pH 7.5 Bound proteins were eluted

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with a gradient of 0–0.4 m NaCl in the same buffer

Inhibi-tory fractions were pooled and subjected to an affinity

col-umn of carboxymethylpapain–Sepharose, prepared as

described previously [5], and equilibrated with 0.02 m Tris–

HCl, pH 7.5, containing 0.3 m NaCl Bound inhibitory

fractions were eluted with 0.02 m NaOH, neutralized with

dilute HCl, and pooled and concentrated by ultrafiltration

(Amicon UM-3)

N-terminal sequence analysis

Automated amino acid sequencing of purified natural and

recombinant macrocypins was performed as described

pre-viously [6] An internal peptide sequence was determined

after cleavage with cyanogen bromide in 80 % formic acid

at room temperature in the dark for 36 h The resulting

peptide fragments were separated using reverse-phase

HPLC, as described previously [6]

Isolation of genomic DNA and total RNA

Basidiocarps of the basidiomycete M procera, harvested

from their natural habitat, were frozen in liquid nitrogen,

homogenized and stored at )80 C until use

High-molecular-weight genomic DNA was isolated from frozen

powdered tissue as described previously [18] and total

RNA was extracted using an RNeasy Kit (Qiagen,

Vienna, Austria) according to the manufacturer’s protocol

for isolation of total RNA from plant tissues and

fila-mentous fungi

Cloning of the genomic and cDNA sequences

encoding macrocypins

First-strand cDNA was synthesized from the total RNA by

RT-PCR using a GeneAmp RNA PCR Core Kit (Applied

Biosystems, Foster City, CA, USA) with anchored

oli-go(dT)-adapter primer (dT(17)3¢RACE) (Table S1) Forward

and reverse degenerate primers (forward 1N-mar-Clit,

nested forward 2N-mar-Clit and reverse C-mar-Clit) were

designed based on the N-terminal amino acid sequence and

an internal peptide fragment sequence First-strand cDNA

was used for PCR to amplify the partial macrocypin cDNA

sequence, which was then used to design specific primers

Forward specific primer (Mp-CliHom-N-uni) was used to

amplify the 3¢ end of the cDNA sequence, using the 3¢

RACE method, together with the 3¢RACE adapter primer

The resulting PCR product was used in a secondary PCR

with two different nested forward primers

(Mp-CliHom-N-A12 and Mp-CliHom-N-A3), together with the 3¢ RACE

adapter primer

To amplify the complete macrocypin gene (mcp) with its

upstream and downstream regions, Genome Walker

libraries were constructed using the Genome Walker

Universal kit (BD Biosciences Clontech) according to the manufacturer’s instructions High-molecular-weight geno-mic DNA (2.5 lg) was digested separately with two restric-tion enzymes (PvuII and StuI) at 37C overnight and after purification by ethanol precipitation, Genome Walker Adaptors were ligated to the digested DNA at 16C over-night The resulting Genome Walker libraries were used as templates in genome walking PCR amplifications, using nested forward specific primers (Mp-CliHom-ter1A and 1) for downstream amplification and nested reverse specific primers (Mp-CliHom-pro 1 and 2) for upstream amplifica-tion, paired with Adaptor Primer 1 and Nested Adaptor Primer 2 provided by the manufacturer Advantage 2 Poly-merase Mix (Clontech) was used for amplification under the conditions suggested by the manufacturer

Complete mcp gene and cDNA sequences were obtained using pairs of primers annealing to the 5¢ UTR (Mcp-N-A12-1 and Mcp-N-A3-1 with nested primers Mcp-N-uni-1

or Mp-CliHom-N-uni) and the 3¢ UTR (Mcp-C-uni-1 with nested primers Mp-CliHom-C-uni, Mp-CliHom-C-A12 or Mp-CliHom-C-A3) regions in two-step PCR amplification, using nested primers in the secondary PCR with recombi-nant Taq polymerase (MBI Fermentas, Vilnius, Lithuania) All PCR products were cloned into the pGEM-T Easy Vector System (Promega, Vienna, Austria) for sequencing

by the Automated DNA Sequencing Service at MWG Biotech (Ebersberg, Germany)

Sequence analyses Sequence analysis and multiple sequence alignments were performed in the BioEdit Sequence Alignment Editor (http://www.mbio.ncsu.edu/bioedit/bioedit.html) Promoter analysis was performed using the Transcription Element Search System (TESS, [19]; http://www.cbil.upenn.edu/cgi-bin/tess/tess) Similarity searches were performed using blastnand tblastn algorithms [13] against different data-bases at the NCBI (http://www.ncbi.nlm.nih.gov/BLAST)

Evolutionary analysis The Datamonkey web interface [20] (http://www.datamon-key.org) was used to examine selective pressure acting upon individual sites of codon alignments Three methods were implemented to test for purifying or diversifying selection: the single likelihood ancestor counting (SLAC); fixed effects likelihood (FEL); and random effects likelihood (REL) [21] A P-value of 0.05 was used for inference of positive and negative selection for individual codons A hierarchical and information theoretic model selection procedure was applied to choose a model of nucleotide substitution HKY85 was selected as the optimal time-reversible nucleo-tide substitution model using the implementation in the HyPhy package [22]

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Expression and purification of recombinant

macrocypins

Whole-length cDNA clones of Mcp1a and Mcp3a were

used as templates in the PCR amplification with Pfu DNA

polymerase (Promega) and primers that introduced NdeI

and BamHI restriction sites to the 5¢ and 3¢ ends of the

insert, respectively After resequencing the resulting

prod-uct, and digesting the inserts and vectors with NdeI⁄ BamHI

(New England Biolabs, Frankfurt am Main, Germany), the

inserts were subcloned into pET3a and pET11a vectors

(Novagen) to generate recombinant proteins without tags

Similarly, NcoI and NdeI restriction sites were introduced

into the 5¢ and 3¢ ends of the whole-length cDNA of

Mcp4a After digestion of the Mcp4a insert and vector with

NcoI⁄ NdeI (New England Biolabs), the insert was

subcl-oned into the pET14b expression vector (Novagen) to

gen-erate protein without tags All constructed expression

vectors with inserts were transformed into E coli

BL21(DE3), which was grown in LB (Luria–Bertani)

med-ium supplemented with 100 lgÆmL)1of ampicillin at 37C

The construct pET14b::Mcp4a was also transformed into

the E coli BL21(DE3) pLysS strain, which was grown at

37C in LB medium supplemented with 100 lgÆmL)1 of

ampicillin and 34 lgÆmL)1 of chloramphenicol When the

attenuance (D) at 600 nm reached 1–1.2, the inducer

iso-propyl thio-b-d-galactoside was added to a final

concentra-tion of 0.4 mm for strains transformed with pET3a or

pET14b constructs and to a final concentration of 1 mm

for strains transformed with the pET11a construct Cells

were then grown for an additional 6 or 8 h They were

har-vested by centrifugation for 15 min at 6000 g, resuspended

in buffer A (50 mm Tris–HCl, pH 7.5, 0.1 % Triton X-100,

2 mm EDTA), frozen and thawed once, then sonicated at

4C The insoluble fraction was separated by

centrifuga-tion for 15 min at 10000 g, redissolved in the same buffer

containing 3 m urea and solubilized by stirring for 4 h at

4C The remaining insoluble material was removed by

centrifugation and the supernatant was applied to a

Sepha-rose S200 column (4· 110 cm) equilibrated with Tris–HCl

buffer, pH 7.5, containing 0.3 m NaCl Inhibitory fractions

were pooled and concentrated by ultrafiltration (Amicon

UM-3) to approximately 1 mgÆmL)1

SDS–PAGE and IEF

Proteins were separated by 12.5% SDS–PAGE under

dena-turing and reducing or non-reducing conditions, as

appro-priate, and stained using Coomassie Brilliant Blue The

molecular mass values of the separated proteins were

estimated using low-molecular-mass standard proteins of

14.4–97 kDa (LMW Calibration Kit; GE Healthcare) The

Phast System (Pharmacia) was used to perform SDS–

PAGE (precast 8–25 % gradient gels) and IEF (precast pH

3–9 gradient gels), as described previously [6] Theoretical

molecular mass and pI values were determined from sequences using the protparam tool available at the Exp-aSy server of the Swiss Institute of Bioinformatics (http:// www.expasy.org/tools/protparam.html)

Inhibition assay Inhibitory activities against papain were determined as described previously [5] using Bz-DL-Arg-2-naphthylamide (Sigma, Taufkirchen, Germany) as the substrate [23]

Enzymes and determination of kinetic constants Human cathepsin H (EC 3.4.22.16) and cathepsin L (EC 3.4.22.15) were purified as described previously [24] Papain (2· crystallized) (EC 3.4.22.2) was further purified by affin-ity chromatography [25] Beta-trypsin (EC 3.4.21.4) was prepared from type IX trypsin (Sigma), as described previ-ously [26] Legumain (EC 3.4.22.34) was isolated from pig kidney cortex following a previously described procedure [27] Recombinant human cathepsin K (EC 3.4.22.38) [28], cathepsin S (EC 3.4.22.27) [29] and cathepsin B (EC 3.4.22.1) [30], all expressed in E coli, were provided by Prof Boris Turk, and recombinant human cathepsin V (EC 3.4.22.43) [31] was provided by Professor Dusˇan Turk (both from the Department of Biochemistry and Molecular and Structural Biology, Jozˇef Stefan Institute, Ljubljana, Slovenia)

Inhibition kinetics for natural and recombinant macro-cypins and clitomacro-cypins were measured under pseudo-first-order conditions, as previously described [6] Continuous kinetic assays were performed for the cysteine proteases papain, cathepsin L and cathepsin V using benzyloxycar-bonyl (Z)-Phe-Arg-7-amido-4-methylcoumarin (AMC) as substrate, and for legumain with Z-Ala-Ala-Asn-AMC as the substrate, while stopped assays were performed for cathepsins K, S and B using Z-Phe-Arg-AMC as the sub-strate and for cathepsin H with Arg-AMC as the subsub-strate [6] Trypsin was assayed using the stopped kinetic assay with fluorogenic substrate Z-Phe-Arg-AMC in 0.1 m Tris– HCl buffer, pH 8.0, containing 1.5 mm EDTA and 0.02 m CaCl2 Data for continuous assays were analyzed by non-linear regression analysis according to Morrison [32], while kinetic constants for cathepsins K, S, B and H, and trypsin, were determined according to Henderson [33] Porcine pepsin (3.4.32.1) from Sigma was assayed in 0.1 m acetate buffer, pH 3.5, using the fluorogenic substrate fluorescein isothiocarbamoyl–hemoglobin, as described for fluorescein isothiocarbamoyl–casein [34]

CD spectroscopy

CD spectra measurements and thermal-unfolding transi-tions were performed on an Aviv model 60

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