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These mutated recombin-ant proteins behave similarly to the wild-type enzyme regarding enzymatic activity, secondary structure, calcium sensitivity and immunoreactivity.. However, immuno

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membrane association, secondary structure and substrate specificity

Vitor Oliveira1, Paula A G Garrido2, Claudia C Rodrigues2, Alison Colquhoun2,

Leandro M Castro2, Paulo C Almeida3, Claudio S Shida3, Maria A Juliano4, Luiz Juliano4,

Antonio C M Camargo5, Stephen Hyslop6, James L Roberts7, Valerie Grum-Tokars8,

Marc J Glucksman8and Emer S Ferro2

1 Laborato´rio de Neurocieˆncias, Universidade da Cidade de Sa˜o Paulo, Brazil

2 Departamento de Biologia Celular e Desenvolvimento, Programa de Biologia Celular, Instituto de Cieˆncias Biome´dicas, Universidade de Sa˜o Paulo, Brazil

3 Universidade de Mogi das Cruzes, Mogi das Cruzes, Brazil

4 Departamento de Biofı´sica, Universidade Federal de Sa˜o Paulo, Brazil

5 Centro de Toxinologia Aplicada (CAT), Instituto Butantan, Brazil

6 Departamento de Farmacologia, Faculdade de Cieˆncias Me´dicas, Universidade Estadual de Campinas, Campinas, Brazil

7 Sam and Ann Barshop Institute for Longevity and Aging Studies, University of Texas Health Science Center, San Antonio, TX, USA

8 Midwest Proteome Center, Department of Biochemistry & Molecular Biology, Rosalind Franklin University of Medicine and Science ⁄ Chicago Medical School, USA

Keywords

calcium; membrane binding; peptide

metabolism; protease; thimet oligopeptidase

Correspondence

E S Ferro, Laborato´rio de Comunicac¸a˜o

Celular, Avenida Prof Lineu Prestes1524

Sala 431, Sa˜o Paulo, 05508-900, SP, Brazil

E-mail: eferro@usp.br

Note

V Oliveira and P.A.G Garrido contributed

equally to this work.

(Received 9 February 2005, revised 24

March 2005, accepted 31 March 2005)

doi:10.1111/j.1742-4658.2005.04692.x

The metalloendopeptidase 24.15 (EP24.15) is ubiquitously present in the extracellular environment as a secreted protein Outside the cell, this enzyme degrades several neuropeptides containing from 5 to 17 amino acids (e.g gonadotropin releasing hormone, bradykinin, opioids and neuro-tensin) The constitutive secretion of EP24.15 from glioma C6 cells was demonstrated to be stimulated linearly by reduced concentrations of extra-cellular calcium In the present report we demonstrate that extraextra-cellular calcium concentration has no effect on the total amount of the extracellular (cell associated + medium) enzyme Indeed, immuno-cytochemical analyses

by confocal and electron microscopy suggested that the absence of calcium favors the enzyme shedding from the plasma membrane into the medium Two putative calcium-binding sites on EP24.15 (D93 and D159) were altered by site-directed mutagenesis to investigate their possible contribu-tion to binding of the enzyme at the cell surface These mutated recombin-ant proteins behave similarly to the wild-type enzyme regarding enzymatic activity, secondary structure, calcium sensitivity and immunoreactivity However, immunocytochemical analyses by confocal microscopy consis-tently show a reduced ability of the D93A mutant to associate with the plasma membrane of glioma C6 cells when compared with the wild-type enzyme These data and the model of the enzyme’s structure as determined

by X-ray diffraction suggest that D93 is located at the enzyme surface and

is consistent with membrane association of EP24.15 Moreover, calcium was also observed to induce a major change in the EP24.15 cleavage site

on distinctive fluorogenic substrates These data suggest that calcium may

be an important modulator of ep24.15 cell function

Abbreviations

EP24.15; metalloendopeptidase 24.15; G6PD, glucose-6-phosphate dehydrogenase; PFA, paraformaldehyde.

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The metalloendopeptidase EC 3.4.24.15 (EP24.15)

rep-resents a distinctive peptide-metabolizing enzyme with

size-selectivity for peptides ranging from 8 to 17 amino

acids [1–3] EP24.15 is a monomeric, soluble, 77-kDa

endopeptidase, thiol-activated, with an isoelectric point

of 5.6 and a pH optimum of activity of 7.4, first

isola-ted from the soluble fraction of the rat brain [4] This

enzyme is distributed in all mammalian tissues so far

examined, with high levels in testis, brain, kidney and

pituitary [5] In the brain, at the electron microscopic

level, EP24.15-like immunoreactivity is observed over

the nuclei, cytoplasm, cross-sectioned dendrites,

myeli-nated and unmyelimyeli-nated axons, axon terminals, and in

both astrocytes and oligodendrocytes [6] Recent

stud-ies have shown that EP24.15 could interact with

peptides generated by the multicatalytic complex

pro-teasome [7,8] Thus, the high concentration of the

enzyme observed inside the cells could be somewhat

related to the degradation of catabolic products

gener-ated by the proteasome [7,9] EP24.15 has been mainly

implicated in extracellular metabolism of

neuropep-tides [5] Supporting an extracellular function of

EP24.15, cell fractionation studies have shown that

EP24.15 is present in a minor form in the particulate

subcellular fractions from the central nervous tissue

[10] and AtT-20 cells [11] Moreover, we previously

reported that EP24.15 is secreted from distinctive cells

lines, such as the rat glioma C6 [12] and mouse AtT20

anterior pituitary [13] Studies in the neuroendocrine

cell line AtT-20 have shown that EP24.15 was enriched

in the regulated secretory pathway [14] and could be

either constitutively released in the extracellular space

or upon stimulation with corticotrophin releasing

hor-mone as well as calcium ionophore A23187 [13]

How-ever, EP24.15 secretion from AtT20 cells is partially

blocked by brefeldin A or nocodazole, suggesting that

this enzyme could be secreted by a pathway different

from that described for other secreted proteins or

neu-ropeptides, such as b-endorphin [13] EP24.15 secretion

from glioma C6 cells is even more unusual, as it is

sti-mulated upon incubation of the cells in the absence of

calcium [12]

We have previously shown that EP24.15 is released

from glioma C6 cells with low calcium concentrations

[12] Using these cells as a model we now show that

extracellular EP24.15 activity (enzyme associated to

cells and present in the medium) was not affected by

the presence or absence of calcium In fact, we have

shown that previously membrane-associated EP24.15 is

shed into the medium in the absence of calcium

Site-directed mutagenesis on a putative calcium binding

motif revealed a role for D93 in EP24.15–membrane

association The location of this residue is on the

surface, and it can bind calcium as corroborated by molecular modeling by the EP24.15 derived X-ray dif-fraction data Moreover, calcium was also observed to affect EP24.15 cleavage site on specific fluorogenic peptides

Results

The rate of shedding of EP24.15 from C6 glioma cells

is inversely proportional to the calcium concentration present in the medium (Fig 1A) To analyze if this increment in EP24.15 activity into the medium was due to secretion, the following experiments were con-ducted: soluble EP24.15 activity was determined in the medium and subtracted from the total activity observed in the presence of cells, the latter correspond-ing to cells + medium activity (Fig 1B) Total extra-cellular EP24.15 activity (cells + media) remains the same with respect to the presence or absence of cal-cium (Fig 1B) However, there is a higher EP24.15 activity in the medium of cells incubated in calcium-free conditions compared with those incubated in calcium-containing medium (Fig 1B) The viability of glioma C6 cells was determined by the Trypan blue dye exclusion method, and suggested that more than 99.5 ± 5% of cells were intact In the same experi-ments, the activity of the cytosolic glucose-6-phosphate dehydrogenase (G6PD), an indicator of cell death, in the medium was < 0.01 ± 0.01 mU per 106 cells (n¼ 5), from a total activity in the whole cell homogenates

of 2.9 ± 0.3 mU per 106 cells (n¼ 5), further suggest-ing that cells were intact after incubation in calcium-free medium

The rabbit anti-EP24.15 serum recognized a single protein band of approximately 78 kDa in the crude homogenate of glioma C6 cells (Fig 2A), suggesting the specificity against this enzyme [15] Importantly, the rabbit anti-EP24.15 antiserum used here has been previously characterized not to show cross-reactivity with the related endopeptidase EP24.16 [6] EP24.15 immunostaining revealed the presence of a diffuse labeling throughout the whole cell that includes an intense nuclear labeling (Fig 2B–E) The intensity of this immunolabeling differed from cell to cell indica-ting a variable expression of EP24.15 in these cells No major differences in the specific immunostaining could

be observed after preincubating the cells in the pres-ence (Fig 2B) or abspres-ence (Fig 2C) of calcium The presence of Triton X100 (0.1%) during the immuno-cytochemical procedures (Fig 2B and C) clearly increases the nuclear labeling for EP24.15, compared

to similar experiments conducted in the absence of the detergent (Fig 2D and E) These data suggest that cell

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permeabilization with detergent is an important issue

regarding the nuclear staining for EP24.15 However,

detergent and calcium removal was not sufficient to

avoid intracellular EP24.15 staining (Fig 2D and E)

Moreover, in control experiments, neither somatic nor

nuclear labeling was observed when the anti-EP24.15

antiserum was preadsorbed with the recombinant EP24.15 (100 lgÆmL)1; Fig 2C)

On the other hand, immunocytochemistry performed with postfixation of the cells in the absence of Triton X100, produces a clear cell-surface immunostaining for EP24.15, with different intensities among the cells (Fig 3A) Under these latter conditions, previous incu-bation of the cells in calcium-free medium strongly affects the intensity of EP24.15 extracellular immuno-staining (Fig 3B) These data suggest the shedding of EP24.15 from the plasma membrane after calcium removal Immunocytochemistry for electron micro-scopy further confirmed the presence of EP24.15 in the extracellular face of the plasma membrane of glioma C6 cells (Fig 4)

We then searched for putative calcium-binding motifs on the surface of EP24.15 that could contribute

to the enzyme’s membrane association At least two possible calcium-binding motifs containing an aspartic acid were identified on these structural analyses (D93 and D159), which were mutated to alanine by site-directed mutagenesis producing the mutants D93A, D159A and the double mutant, D93⁄ 159A The corres-ponding recombinant proteins were expressed in bac-teria and purified to homogeneity (Fig 5A) Western blot analyses show that such point mutations have no effect on antiserum recognition (Fig 5B) The follow-ing experiments were performed in an attempt to corre-late a possible effect of the above point mutations with the calcium-dependent EP24.15 membrane association The homogeneously purified EP24.15 recombinant pro-teins (wild-type or mutant) were incubated with glioma C6 cells and the putative membrane binding was ana-lyzed by confocal microscopy Incubation of either wild-type EP24.15 or the D159A mutant with the glioma C6 cells produced a clear increment in the immunoreactivity of EP24.15 associated with the extra-cellular surface of these cells (Fig 6A and B) when compared with control performed without adding any

of the recombinant EP24.15 (Fig 6E) On the other hand, incubation of the glioma C6 cells with either D93A or D93⁄ 159A mutated proteins was not able to produce a similar increment in the extracellular immu-noreactivity related to EP24.15 (Fig 6C and D) Con-trol experiments under these conditions show that without the addition of EP24.15 the basal immunoreac-tivity was greatly reduced (Fig 6E) Taken together, these data suggest the D93 residue of EP24.15 within the putative calcium-binding motif 89SPNKD93 is a possible mediator of enzyme–membrane association

To examine a structure–function correlate of the D93–membrane association and the location of the resi-due within the three-dimensional realm of the enzyme,

Fig 1 Calcium-dependent EP24.15 released into the medium.

EP24.15 activity in intact cells was determined using

b-lipotro-pin(61–69) (YGGFMTSEK; 100 l M ) as substrate b-lipotropin(61–69)

fragments were separated by reverse phase HPLC and the

EP24.15 activity was based on the generation of [Met5]enkephalin

calculated by comparing the peak areas of each of those products

with the corresponding peak area of the same synthetic peptides

of known concentrations The EP24.15 enzymatic activity released

into the medium was determined fluorimetrically using the

substrate Abz-GGFLRRV-EDDnp To discern peptidolytic activity

exclusively due to EP24.15, the inhibitors

N-(1-(R,S)-carboxyl-2-phenylethyl)-AAF-p-amino-benzoate (CFP-AAF-pAB) and ⁄ or the

dipeptide Pro–Ile were used All enzymatic determinations were

conducted under linear conditions where product formation was

directly proportional to enzyme concentration, and obeys zero-order

kinetic parameters with < 10% of total substrate consumed during

the course of the assay Results are means of three independent

determinations ± SE (A) EP24.15 release into the glioma C6 cells’

medium is inversely proportional to the extracellular calcium

con-centration (B) Total extracellular EP24.15 activity is not affected by

calcium, however, the amount of enzyme that is released in the

medium is largely increased after incubating the glioma C6 cells in

medium that lacks calcium.

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a hypothetical intermolecular interaction using calcium

as a ligand was performed (Fig 7) The calcium is

modeled in green and is hydrogen bonded to two

amino acids The first amino acid is D93, the object of

study in this work Using an ensemble of coordinates

of well studied Asp–calcium interactions, the carboxy

groups of the Asp are within hydrogen bonding

dis-tance (2.5–3 A) for calcium The coordinates for Fig 7

were obtained from the crystal structure of thimet

oligopeptidase deposited at the Research Collaboratory

for Structural Bioinformatics (Protein Data Bank ID

#1S4B) The figure was drawn with the computer modeling program spock and then subsequently ren-dered using raster 3d [16] To ensure that electro-statics were properly accounted for, calculations were performed and slightly negative potentials were obtained for the D93

Furthermore, we investigated the calcium effects on EP24.15 secondary structure, enzymatic activity and substrate specificity Enzymatic activity of the mutated

Fig 2 EP24.15 immunoreactivity in glioma C6 cells EP24.15 presence in glioma C6 cells was further confirmed by immunochemical reac-tions (A) The antiserum raised against the rat testis recombinant EP24.15 recognized in the whole homogenate of glioma C6 cells a single protein band corresponding to the expected Mrof EP24.15 For details see Experimental procedures (B–E) Prior to each immunocytochem-istry experiment the DMEM culture medium was removed and cells were rinsed three times with fresh NaCl ⁄ P i , with (B, D) or without (C, E) 1.2 m M of calcium, and incubated for 30 min in 4% formaldehyde, pH 7.4 After three cycles of NaCl ⁄ P i washing (15 min each) cells were incubated for 2 h in the presence of rabbit anti-EP24.15 serum (1 : 4000) diluted in NaCl ⁄ P i containing 3% normal goat serum, 1% BSA, in the presence (B, C) or absence (D, E) of 0.1% of Triton X100 Labeled cells were examined under a Zeiss laser confocal microscope (CLSM 410) equipped with an Axiovert 100 inverted microscope and an Argon ⁄ Krypton laser Cy3-tagged molecules were excited at a wave-length of 568 nm Images were acquired sequentially as single transcellular optical sections and averaged over 32 scans per frame They were then processed using the Carl Zeiss CLSM software (version 3.1) and stored on Jazz disks for further retrieval and editing Final com-posites were prepared using Adobe PHOTOSHOP without modifying the spectral characteristics of the original signal Control experiments were performed as described; no specific cell labeling could be observed (data not shown) The data presented are representative of three experi-ments performed independently and exhibiting similar results.

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proteins either in the presence or absence of calcium

was not different from the wild-type EP24.15

(Fig 5C) The superimposable CD spectra indicates

that there are no gross perturbations in expression of the mutant proteins with respect to folding CD spec-tra of wild-type EP24.15 and mutants without

Fig 4 Extracellular EP24.15 immunogold labeling observed by electron microscopy Cells incubated in the presence of calcium were processed for electron microscopy using a pre-embedding procedure as des-cribed (A) Lower magnification of an entire glioma C6 cell observed in the electron microscope and immunolabeled at the extracellular surface for EP24.15 (arrows) (B) Higher magnification of the cell shown

in (A) immunolabeled for EP24.15 (arrows) Scale bar: A, 140 nm; B, 500 nm.

Fig 3 The effects of calcium on the extracellular EP24.15 immunoreactivity in glioma C6 cells (A, B) To observe exclusively the extracellular EP24.15 immunoreactivity in glioma C6 cells, cells were incubated for 15 min at 4 C in the presence of rabbit anti-EP24.15 serum (1 : 500), diluted in NaCl ⁄ P i containing 3% normal goat serum and 1% BSA, prior to fixation with 4% formaldehyde pH 7.4 Labeled cells were exam-ined under a Zeiss laser confocal microscope (CLSM 410) equipped with an Axiovert 100 inverted microscope and an Argon ⁄ Krypton laser Cy3-tagged molecules were excited at a wavelength of 568 nm Images were acquired sequentially as single transcellular optical sections and averaged over 32 scans per frame They were then processed using the Carl Zeiss CLSM software (version 3.1) and stored on Jazz disks for further retrieval and editing Final composites were prepared using Adobe PHOTOSHOP without modifying the spectral characteristics of the original signal Immunocytochemistry reactions were conducted after incubation of the glioma C6 cells in the presence (A) or absence (B) of calcium for 1 h Note that cells previously incubated in medium lacking calcium (B) show a reduced extracellular labeling for EP24.15 compared to cells incubated in the presence of calcium (A) The intensity of the immunostaining varies from cell to cell but is consistently stronger in cells previously incubated in the presence of calcium compared to those incubated in the absence of calcium (1) Confocal regu-lar image; (2) nomarski; (3) glow scale The immunolabeling intensity is shown in the graphic (intensity grows from green to red) No immu-nolabeling is observed after preabsorbing the anti-EP24.15 serum with recombinant EP24.15 (100 lgÆmL)1; data not shown).

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smoothing and curve fitting shows a predominance of

a-helical structures (Table 1; Fig 8A–D), which is

similar to the estimated consensus secondary structure

prediction obtained from different algorithms (http://

npsa-pbil.ibcp.fr), and more importantly, the structure

of the protein solved by X-ray crystallography [17]

Far UV-CD analysis indicated that the small

differ-ences observed among the wild-type and mutated

EP24.15 falls within the experimental error determined

using three different preparations of the enzyme (data

not shown) The inter-experimental errors are  4%

However, calcium (2.2 mm) addition reduced the

a-helical content with a concomitant increase in the

b-strand, turn and unordered structure of EP24.15

(Table 1); similar results were observed in the near UV-CD spectra, collected either in the presence or in the absence of calcium (data not shown) These data clearly show that calcium affects EP24.15 secondary structure The D93A mutation alone or in combina-tion with D159A that prevents EP24.15 membrane association seems insufficient to completely prevent the calcium changes induced in the enzyme secondary structure (Table 1), suggesting that structural changes may not correlate entirely with the enzyme membrane association

With regards to substrate cleavage specificity, the gradual increase in the calcium concentration from

0 to 50 mm raised the preference of the wild-type EP24.15 for the cleavage at the R–R bond in the ortho-aminobenzoic acid (Abz)-GGFLRRVQ-EDDnp substrate from 64% (0 mm Ca+2), to 79% (50 mm

Ca+2) with a corresponding decrease in the cleavage at the L–R peptide bond from 36% (0 mm Ca+2) to 21% (50 mm Ca+2; Table 2) This variation is even more pronounced with the Abz-GGFLRRDQ-EDDnp sub-strate, which yielded cleavages of 63% and 37% at the L–R and R–R bonds, respectively, in the absence of

Ca2+ In the presence of 50 mm of Ca2+the preferred hydrolyzed peptide bond changes to 26% and 74% for the cleavage at the L–R and R–R bonds, respectively (Table 2) Moreover, after a 2 mm calcium incre-ment, the kcat⁄ Km (lm)1Æs)1) ratio of both Abz-GGFLRRVQ-EDDnp and Abz-GGFLRRDQ-EDDnp started to change (Table 2) These data suggest that in the presence of high calcium concentrations, as found

in specific cellular microenvironments [18], EP24.15 substrate specificity could be changed Mutations of EP24.15 either at D93 or D159 have no major effects

on the ratio of Abz-GGFLRRDQ-EDDnp peptide bond cleavage after calcium addition (data not shown)

Discussion

The most important finding of this report is that cal-cium regulates the membrane association, secondary structure and substrate specificity of the endo-oligo-peptidase EP24.15 Site-directed mutagenesis indicated that residue D93 plays an important role in this cal-cium-dependent membrane association of EP24.15 in glioma C6 cells (Fig 6) This finding is consistent with the position of the residue on the surface of the enzyme as modeled on the X-ray structure (Fig 7), although we have been unable to demonstrate the direct interaction of calcium with the enzyme More-over, biochemical analyses also indicated that calcium affects EP24.15 secondary structure (Fig 8) and

A

4

- Ca2+

+ Ca2+

3

2

1

0

EP24.15

78kDa

78kDa

B

C

Fig 5 SDS ⁄ PAGE and western blot of EP24.15 recombinant

pro-teins (A) SDS ⁄ PAGE stained with Comassie blue shows the

homo-geneity of the recombinant proteins (5 lgÆlane)1) EP24.15 wild-type

(WT), D93A (93), D159A (159) and D93 ⁄ 159A (93 ⁄ 159) after the

affinity purification as described (B) Western blotting of the

recom-binant proteins (0.5 lgÆlane)1) EP24.15 wild type (WT), D93A (93),

D159A (159) and D93 ⁄ 159A (93 ⁄ 159) using the anti-EP24.15

serum Note that none of the mutations affected the specificity of

the antiserum for the EP24.15 (C) Comparative enzymatic activity

of EP24.15 WT and mutants D93A, D159A and D93 ⁄ 159A

meas-ured with the fluorogenic substrate QFS in the absence and in the

presence of 2 m M CaCl2 Note that calcium slightly increases

EP24.15 enzymatic activity in these assays.

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Fig 6 Immunocytochemical staining of glioma C6 cells after incubation with the recombinant EP24.15 proteins Glioma C6 cells were prein-cubated at 4 C in the presence (A–D) or absence (E) of 2 lg of either EP24.15 wild-type (A), D159A (B), D93A (C) or D93 ⁄ 159A (D), for

30 min, in DMEM containing BSA 0.05% The medium was then removed and cells were rinsed three times with DMEM and incubated at

4 C for 30 min in the presence of the anti-EP24.15 serum (1 : 500) The excess of primary antiserum was removed rinsing the cells three times in 1 mL of NaCl ⁄ P i containing 5% BSA, before fixing the cells in PFA 4% The secondary anti-(rabbit Cy3) Ig (1 : 250; Sigma) was used to develop the immunoreaction Note that the intensity of the EP24.15-related immunostaining is reduced for either the D93A or D93 ⁄ 159A proteins compared to the wild-type and D159A.

Fig 7 Molecular modeling of the surface EP24.15 D93 residue complexed with a calcium ion On the left a diagram of the side chain of D93 and a bound calcium (gray sphere) mapped onto the surface of the recently solved EP24.15 structure ([40] PDB ID #1S4B) On the right

is a magnified view focusing on the D93 side chain and interaction with calcium The EP24.15 model with the calcium ion was energy mini-mized using Molecular Operating Environment software, and the figure was generated using SPOCK

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changes the enzyme substrate specificity (Table 2).

These data suggest that calcium is an important

medi-ator of EP24.15 biological function

It has been previously shown that in the brain

EP24.15 is present both in neurons and glial cells [5,6]

Glial cells are at least 10 times more abundant than

neurons in the brain [19] and during the past years

they have been recognized to be more than a

connect-ive tissue responsible for neuronal support [20] Glial

cells are now identified as excitable cells capable of modulating neuronal stimulus [19] As for neurons, glial cells can also produce growth factors, pro-hor-mone processing enzymes [21], receptors [22], neuro-transmitters [19] and neuropeptides [23] However, in contrast to neurons, which process peptides within the secretory pathway, glial cells secrete immature peptide precursors [24,25] Glial post-translational processing

of opioid peptides precursor (e.g proenkephalin) is therefore thought to occur in the extracellular environ-ment, as demonstrated by the presence of protease inhibitors in the cultured medium of striatal astrocytes that prevented Met-enkephalin generation from secre-ted proenkephalin [25–27] Therefore, the present results suggest that in physiological conditions EP24.15 would be able to associate with the extracellu-lar surface of glial cells and could participate in the metabolism of a specific set of peptides released by these cells and⁄ or from neurons Thus, further charac-terization of peptide-processing enzymes such as EP24.15 in the extracellular milieu of glial cells is of neurobiological significance

Table 1 Calcium effects on the secondary structure of EP24.15.

TBS

TBS + 2.2 m M Ca 2+

Fig 8 Far UV-CD spectra of the EP24.15 WT (A), D93A (B), D159A (C) and D93 ⁄ 159A (D) mutants both in the absence (n) or in the pres-ence of CaCl 2 2 m M(h) Note that calcium addition reduces the a-helical content of all EP24.15s analyzed (Table 1).

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In a previous study we reported that EP24.15

secre-tion from glioma C6 cells was increased linearly by

reduced extracellular calcium concentrations [12]

These data were paradoxical to many other secretory

systems in which extracellular calcium is required for

secretion [28]; therefore, the mechanism behind this

calcium effect on EP24.15 secretion was investigated

further It is known that extracellular calcium is not

required for constitutive protein secretion [29] and

reduces the release of parathyroid hormone [28]

Bio-chemical observations shown here indicated that

extracellular calcium concentrations could not change

the overall rate of peptide metabolism from intact

gli-oma C6 cells in culture (Fig 1) These data suggest

that the overall extracellular EP24.15 enzymatic

activ-ity was not affected by altered calcium

concentra-tions Consequently, we conclude that calcium

removal was not really affecting EP24.15 secretion

from glioma C6 cells Another possibility was that

low extracellular calcium concentration compromised

the EP24.15 association with the E face of the plasma

membrane The presence of EP24.15 associated with

the E face of the plasma membrane in glioma C6

cells was confirmed by immunocytochemistry

experi-ments both at the light and electron microscopy

levels Indeed, immunocytochemistry analyses shown

here suggested that EP24.15 extracellular

immuno-reactivity is deeply affected when extracellular calcium

is removed from glioma C6 cells Thus, it seems clear

that a deficiency in extracellular calcium affects the

shedding of EP24.15 from the plasma membrane of

glioma C6 cells into the medium, suggesting that

EP24.15–membrane association within cells can be

dynamically modulated While further investigation

would be necessary, this mechanism could be relevant for the intracellular traffic and secretion of EP24.15 [13,30], since it could dynamically locate the enzyme

in specific cell membranes, such as the endoplasmic reticulum [6], Golgi apparatus, secretory vesicles [14] and plasma membrane [11]

Interestingly, EP24.15 contains neither a leader pep-tide sequence nor any other hydrophobic domains to mediate its interaction with biological membranes [31] and the mechanism responsible for the enzyme mem-brane association in neuroendocrine cells remains unknown [6,11] The EP24.15 crystal structure was recently solved [17] showing that the enzyme surface contains 80% of all charged residues Among these amino acid residues D93 and D159 were investigated

as possible mediators of EP24.15–membrane associ-ation The D93 amino acid residue is located within the EP24.15 89SPNKD93 sequence, which recalls the calcium-binding motif DXSXS previously described [32] On the other hand, D159 residue is the EP24.15 corresponding metal divalent-binding site residue iden-tified in the homologue endopeptidase EP24.16; both D93 and D159 residues are located at the external sur-face of the enzyme [17,33] The D93A or D93⁄ 159A double mutations were shown to affect the calcium-dependent association of EP24.15 to the plasma membrane of glioma C6 cells, while the D159A point mutation had no such effect The molecular modeling

of the interaction of calcium and D93 on the surface

of the enzyme has the correct electrostatic potential Additionally, the calcium is far enough from the active site zinc that no direct interaction can be postulated for direct effects on catalysis Changes appear to be transduced through changes in secondary structure Circular dichroism experiments reported here clearly shows that calcium affects EP24.15 secondary struc-ture, reducing the a-helical content by 3–8% It is well known that calcium affects the conformational struc-ture of several proteins [34–37], and that could provide

a possible mechanistic explanation for the data shown above The D93A or D93⁄ 159A mutations that strongly affected EP24.15 membrane association were not sufficient to prevent the secondary structure altera-tions of EP24.15 induced by calcium Hence, it is plausible to suggest that the structural changes induced

by calcium on EP24.15 secondary structure are not correlated with the membrane association Therefore, the exact role of D93 in EP24.15 membrane associ-ation remains unknown A possible explanassoci-ation that needs further attention is that D93 mediates the inter-action of EP24.15 with a yet unidentified membrane protein Molecular modeling corroborates that the resi-due is on the surface

Table 2 Influence of calcium concentration on EP24.15 peptide

bond cleavage.

[Ca 2+ ]

(m M )

Cleaved bond (%)

Abz-GGFLRRVQ-EDDnp Abz-GGFLRRDQ-EDDnp

L–Rb R–Rb

k cat ⁄ K ma

(l M )1.s)1) L–Rb

R–Rb

k cat ⁄ K ma

(l M )1.s)1)

a The SD were < 5% for any of the obtained kinetic parameters b In

three independent assays conduced in triplicate the errors obtained

in the percentage of hydrolyzed peptide bond was less than 3%.

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A previous report demonstrated the influence of

cal-cium and other divalent cations activating EP24.15

enzyme activity, in a substrate dependent manner [38]

consistent with our data Another recent study has also

shown the influence of calcium on the acid limb of a

pH-dependence activity curve of EP24.15 [39]

Fluoro-genic substrates have also been used previously to

show the influence of different salts on the EP24.15

activity [40] Our present data confirm these previous

studies as calcium affected the kcat⁄ KMratio of at least

two fluorogenic substrates hydrolyzed by EP24.15

(Table 2) In addition, we have shown here that

calcium affect the ratio of peptide bond (L–R⁄ R–R)

cleavage observed with the fluorogenic substrates

Abz-GGFLRRDQ-EDDnp and Abz-GGFLRRVQ-EDDnp

assayed These effects were more pronounced using the

substrate Abz-GGFLRRDQ-EDDnp, which contains

an aspartic acid residue that could interact directly

with calcium However, the effect was also verified

with the substrate Abz-GGFLRRVQ-EDDnp that

does not contain any negatively charged residues to

bind calcium directly, thus suggesting that the

struc-tural changes induced by calcium on EP24.15

secon-dary structure could modulate its substrate specificity,

even altering the cleavage site on specific peptides

Moreover, the results obtained with the substrate

Abz-GGFLRRDQ-EDDnp seem to indicate the presence

of a positive residue at the S3¢ subsite Thus, if the

kcat⁄ KMratio obtained from the kinetics with the

sub-strate Abz-GGFLRRDQ-EDDnp containing a

negat-ive change at P3¢ is decomposed into the cleavage sites,

it is possible to verify that the cleavage at the R–R

bond was activated while the cleavage at the L–R

bond was inhibited at higher calcium

concentra-tions Similar analyses using the substrate

Abz-GGFLRRVQ-EDDnp, which that does not contain a

negative change at P3¢, shows inhibition of both R–R

and L–R cleaved sites These observations suggest that

EP24.15 contains a positively charged residue at the

S3¢ subsite This is in agreement with previously

pub-lished reports [3,40], and could be of importance in the

development of new specific substrates and⁄ or

inhibi-tors for the enzyme, as the detailed architecture of the

enzyme and its interactions with substrates are

deci-phered

The mutations D93A and D159A neither prevented

the effect of calcium ions on the cleaved peptide

bond ratio (L–R⁄ R–R) nor changed the kcat⁄ KM

ratio for these substrates (data not shown) These

mutations that could not prevent the structural

chan-ges induced by calcium on EP24.15 structure as

observed with the far UV-CD assays suggest the

possible existence of other calcium binding site(s) in

the EP24.15 structure, which have not yet been des-cribed

In conclusion, the present report provides evidence that EP24.15 is able to associate to the extracellular face of the plasma membrane in a calcium dependent fashion Calcium was also shown to change the secon-dary structure by reduction of a-helical content and to change the substrate specificity of EP24.15 Together, these new findings are useful to understand the com-plex organization of peptide metabolism within cells mediated by divalent cation by altering and regulating enzyme localization within subcellular compartments

in the cell

Experimental procedures

Cell culture Glioma C6 cells were cultured in Dulbecco’s modified Eagle’s medium (DMEM; Invitrogen, Carlsbad, CA, USA)

previ-ously described [12] The cells were maintained in 12-well

air Prior to the experiments, the culture medium from 60– 80% confluent cells was removed and cells were rinsed three times with 1 mL Henk’s buffer (136 mm NaCl,

cal-cium-free experiments, cells were previously incubated for

Determination of EP24.15 activity of intact glioma C6 cells by HPLC

Total extracellular EP24.15 activity was determined using

hydrolysis was consistently < 10% during all the determi-nations The enzymatic reactions were terminated by

by reverse phase HPLC using a C18 lBondapak column

reaction was evaluated by determining the amount of

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