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This enzyme, TcrPDEC, is a member of the class I PDEs, as determined from the presence of a characteristic signature sequence and from the conservation of a number of functionally import

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phosphodiesterase from Trypanosoma cruzi

Stefan Kunz, Michael Oberholzer and Thomas Seebeck

Institute of Cell Biology, University of Bern, Bern, Switzerland

The cell biology of Trypanosoma cruzi, the causative

agent of South American Chagas’ disease, has been

extensively studied Surprisingly, still very little is

known about the role of cyclic nucleotide signaling in

this organism [1] A number of earlier studies have

indicated a role of cAMP in differentiation [2,3], and

the existence of a nitric oxide regulated guanylyl

cyclase has been suggested [4] In T cruzi

epimasti-gotes, a cAMP-regulated transcript has been identified

that can be induced by elevated cAMP levels [5] The

corresponding gene, TC26, was later found to code for

an RNaseH and to be localized on a large family of

repetitive genetic elements More recently, the T cruzi

genome was shown to code for several adenylyl

cyclas-es, all predicted to be similarly organized, consisting of

a large N-terminal, presumably extracellular region, which is followed by a single transmembrane helix and

a C-terminal catalytic domain [6] The structure of these cyclases is entirely different from that of their mammalian counterparts, but closely similar to that of the cyclases characterized in Leishmania donovani [7] and in African trypanosomes [8–10] One of these adenylyl cyclases, TczAC, was found to interact with a paraflagellar rod protein, and is most likely located in the flagellum [11]

A cAMP-specific phosphodiesterase (PDE) activity has been demonstrated in T cruzi by various laborat-ories [12,13] Recently, the first cyclic-nucleotide-specific PDE from T cruzi has been identified and characterized at the molecular level [14] This enzyme,

Keywords:

Chagas’ disease; cyclic nucleotides; FYVE

domain; kinetoplastids; phosphodiesterase

Correspondence

T Seebeck, Institute of Cell Biology,

Baltzerstrasse 4, CH-3012 BERN,

Switzerland

Fax: +41 31 6314684

Tel: +41 31 6314649

E-mail: thomas.seebeck@izb.unibe.ch

Website: http://www.izb.unibe.ch

Nucleotide sequence data have been

sub-mitted to the DDBJ ⁄ EMBL ⁄ GenBank

data-bases under the accession numbers

AJ889575 and AJ889576 for TcrPDEC

alle-les 1 and 2, respectively.

(Received 26 August 2005, revised 20

Octo-ber 2005, accepted 27 OctoOcto-ber 2005)

doi:10.1111/j.1742-4658.2005.05039.x

Cyclic-nucleotide-specific phosphodiesterases (PDEs) are key players in the intracellular signaling pathways of the important human pathogen Trypano-soma cruzi We report herein the identification of an unusual PDE from this protozoal organism This enzyme, TcrPDEC, is a member of the class

I PDEs, as determined from the presence of a characteristic signature sequence and from the conservation of a number of functionally important amino acid residues within its catalytic domain Class I PDEs include a large number of PDEs from eukaryotes, among them all 11 human PDE families Unusually for an enzyme of this class, TcrPDEC contains a FYVE-type domain in its N-terminal region, followed by two closely spaced coiled-coil domains Its catalytic domain is located in the middle of the polypeptide chain, whereas all other class I enzymes contain their cata-lytic domains in their C-terminal parts TcrPDEC can complement a PDE-deficient yeast strain Unexpectedly for a kinetoplastid PDE, TcrPDEC is a dual-specificity PDE that accepts both cAMP and cGMP as its substrates

Abbreviations

DMSO, dimethyl sulfoxide; EHNA, erythro-9-(2-hydroxy-3-nonyl)adenosin; FYVE, domain containing Fab1p, YOTB, Vac1p and EEA1PDE; GST, glutathione-S-transferase; IBMX, isobutyl methyl xanthine; LmPDEC, phosphodiesterase from Leishmania major; PtdIns(3)P, phosphatidyl inositol-3-phosphate; TbPDEC, phosphodiesterase from Trypanosoma brucei; TcrPDEC, phosphodiesterase from Trypanosoma cruzi.

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TcPDE1 is entirely cAMP-specific, and it is located

along the flagellum In terms of its amino acid

sequence, TcPDE1 is a close homologue of the

Trypano-soma bruceiPDEs TbPDE2B [15] and TbPDE2C [16],

as well as of LmPDEB1 and LmPDEB2 of Leishmania

major (Johner et al., unpublished results) All of these

enzymes belong to the class I PDEs [17] TcPDE1

con-tains two GAF domains [18,19] in its N-terminal

moi-ety, and a C-terminal catalytic domain TcPDE1, as all

its other kinetoplastid homologues, is highly

cAMP-selective

This study reports the identification and

characteri-zation of a novel and rather unusual PDE from T cruzi

According to the recently proposed unifying

nomen-clature for kinetoplastid PDEs [20], this enzyme was

designated as TcrPDEC Based on the amino acid

sequence of its catalytic domain, TcrPDEC

unambigu-ously belongs to the class I PDEs However, it is a

rather unusual PDE in several respects: (a) unlike all

other class I PDEs, its catalytic domain is localized in

the middle of the polypeptide chain, and not at its

C-terminus; (b) the N-terminal region of TcrPDEC

contains a FYVE-type domain [21,22], a functional

domain that has not been found in any PDE so far;

and (c) TcrPDEC is the first dual-substrate PDE, with

similar Kmvalues for cAMP and cGMP, that has been

identified in kinetoplastids

Results

Identification of TcrPDEC

When the T cruzi database (http://www.genedb.org/

genedb/tcruzi) was screened for putative PDEs, a gene

was identified that codes for a rather unusual PDE,

TcrPDEC (temporary gene identification number

Tc00.1047053506697.20) The open reading frame of

TcrPDEC was amplified from genomic DNA, and

sev-eral PCR products were sequenced This analysis

revealed the presence of two distinct alleles which

dif-fer by 62 bp (out of the 2775 bp of the entire open

reading frame; 2.2% sequence divergence) These single

nucleotide polymorphisms translate into 38 amino acid

changes (4.1% amino acid substitutions; 21 conserved,

17 nonconserved) Only six of these substitutions occur

in the catalytic domain of the enzyme, and none of

them affects a residue that is crucial for function (see

below) Southern blot analysis of T cruzi genomic

DNA by hybridization with the complete open reading

frame of TcrPDEC results in restriction enzyme

pat-terns that are compatible with the nucleotide sequence

of TcrPDEC, demonstrating that it represents a single

copy gene (Fig 1) EcoRI, PstI and EcoRV cut once

within the open reading frame, BamHI does not cut, and HindIII cuts three times, resulting in two frag-ments too small to be detected by hybridization and one fragment of two kilobases The hybridization of SalI-digested DNA confirmed the polymorphism of one SalI site detected by the sequence analysis of the two alleles The establishment of TcrPDEC as a single-copy gene is not entirely trivial, as large parts of the

T cruzigenome have undergone a duplication [23]

Functional domains of TcrPDEC The open reading frame of TcrPDEC codes for a pro-tein of 924 amino acids (calculated relative molecular mass of 103 169, calculated pI¼ 5.91) with several functional domains (Fig 2A) The N-terminus (P10–

G73) contains a FYVE-type domain, an acronym composed of the designations of the first four repre-sentatives Fab1p, YOTB, Vac1p and EEA1 [21], that is followed by two closely spaced coiled-coil regions (D144–D179 and K207–E264) FYVE-type domains are zinc-finger-like structures, which are currently divided

10 8 6 5 4

3

2 1.5

1

0.5

EcoRI SalI EcoRV BamHI PstI HindIII

kb

Fig 1 TcrPDEC is a single copy gene Genomic DNA of T cruzi hybridized with a probe representing the entire open reading frame

of TcrPDEC.

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into two classes: The classical FYVE domains

(exam-ples: (HsEEA1, accession number Q15075; DmHRS,

accession number Q960· 8; and ScVps27p, accession

number P40343) share three consensus motifs (WXXD,

R + HHCR and RVC), and they bind specifically

to

membrane-embedded-phosphatidyl-inositol-3-phos-phate [PtdIns(3)P] FYVE-variant domains such as

HsDFCP1 and AtPRAF1 lack some of the conserved

residues (Fig 2B), but still bind PtdIns(3)P, albeit with

lower affinity The FYVE-related domains (e.g

rabphi-lin 3 A (P47709) or human Rim1 (Q86UR5) exhibit

a still higher sequence divergence in the consensus

region Their function is still undetermined The

align-ment of the FYVE-type domain of TcrPDEC places it

close to the FYVE-variant domains All eight cysteine

residues predicted to be involved in Zn2+-binding are

fully conserved (Fig 2C), as are the two predicted

heli-cal regions The two hydrophobic amino acids that are

inserted in the membrane upon PtdIns(3)P binding

(L185 and L186 in ScVps27p [22]); are represented by

L30 and F31 of TcrPDEC When matched with the

WxxD R + HHCR RVC motif of FYVE domains, the sequence of TcrPDEC exhibits several alterations

A glutamate residue at position four of the first block

is substituted by aspartate, arginine at the beginning of the second block is replaced by an alanine, the two adjacent histidine residues are replaced by serine and glutamine, the subsequent arginine is replaced by pro-line, and finally the arginine of the third block is replaced by a lysine The consequence of these replace-ments is a decrease of the overall net charge of the motive from +4 to +1 The effect of these changes

on a putative membrane binding of the FYVE-type domain of TcrPDEC remains to be explored, but they render the TcrPDEC domain unlikely to bind to PtdIns(3)P This prediction is confirmed by the obser-vation that the recombinant FYVE domain of TcrPDEC does not bind to PtdIns(3)P, nor to PtdIns(3,4)P2, PtdIns(4,5)P2, PtdIns(3,4,5)P3, phos-phatidic acid, phosphatidyl choline, phosphatidyl ser-ine or phosphatidyl inositol in a dot-spot assay [24,25] (data not shown)

FYVE DmHrs

PHD HsKAP-1

FYVE-related HsRIM1

FYVE-related RnRPH3A

TcPDEC

FYVE-variant AtPRAF1

FYVE-variant HsDCFP1

FYVE ScVps27p

FYVE HsEEA1

C

Fig 2 The FYVE-type domain of TcrPDEC (A) Functional organization of TcrPDEC: 1, FYVE-type domain; 2 and 3, coiled-coil regions; 4, cata-lytic domain (B) Dendrogram of FYVE domains: TcrPDEC; HsEEA1, human early endosome antigen 1 (accession number Q15075); DmHrs, Drosophila Hrs (Q960 · 8); ScVps27p, S cerevisiae vacuolar sorting protein (P40343); HsDFCP1, human double FYVE-containing protein 1 (Q9HBF4); AtPRAF1, Arabidopsis PRAF1 (Q947D2); RnRPH3A, rat rabphilin-3 A (P47709); HsRIM1, human Rim1 (Q86UR5) (C) Alignment of FYVE and FYVE-related domains Grey boxes: the conserved Zn 2+ coordinating cysteine residues The FYVE domain signature motifs WxxD,

R + HHCRxCG and RVC are given in bold, underlined letters Box 1: turret loop [41]; box 2: a-helix found in the structures of HsEEA1, DmHrs, ScVps27p and RnRabphilin-3 A Horizontal box: putative dimer interface of EEA1 [42].

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Downstream of the FYVE domain, TcrPDEC is

pre-dicted to contain two closely spaced coiled-coil regions

(S150–L174and K207–D264) These might serve to

dimer-ize the FYVE domains in a way similar to the

struc-ture that was determined for EEA1 [26]

To explore if these regions are indeed essential for

stabilizing the FYVE domain in the dimeric state, the

FYVE domain was expressed either alone (amino acids

1–74 of TcrPDEC), or in conjunction with the

coiled-coil region (amino acids 1–272) Gel filtration analysis

demonstrated that already the FYVE domain alone

migrates as a stable dimer (calculated molecular mass

8.2 kDa; apparent molecular mass upon gel filtration:

16.2 kDa) (Fig 3A) The construct containing the two

coiled-coil regions in addition to the FYVE domain (calculated molecular mass 30.5 kDa) eluted with an apparent mass of 199.7 kDa, indicating the formation

of a higher order complex (Fig 3B)

In TcrPDEC, the catalytic domain is located in the middle of the polypeptide chain of 924 amino acids (T291–S657; Fig 2A) This is very unusual for a class I PDE, as all other members of this PDE class contain the catalytic domain in their C-terminal portions Nev-ertheless, the catalytic domain of TcrPDEC unambigu-ously identifies it as a class I PDE (Fig 4) This PDE class includes all 11 human PDE families, and all

of its members contain the signature motif HD(LIV-MFY)xHx(AG)xxNx(LIVMFY) Their catalytic domains share 30–40% amino acid sequence identity between families [17] The overall sequence of the TcPDE-FYVE catalytic domain conforms well with that of other class I PDEs It shares between 24 and 33% of amino acid sequence identity with the 11 human PDE families, and 45 and 57% sequence iden-tity with its putative orthologs in L major (lmjPDEC) and T brucei (TbrPDEC; unpublished data) All resi-dues that have been identified as important for sub-strate recognition, selectivity and catalysis in the human PDEs [27,28] are conserved in TcrPDEC with respect to HsPDE4B2 (Fig 4) The two metal binding sites are predicted to be formed by residues H368, H372,

H409, E410, His413, N418, L438, D439, E481, M482 and

E521[28] The hydrophobic pocket that accommodates the purine moiety of the substrate is also conserved and predicted to consist of Y367, I522, A524, S525, A532,

W535, L536, I538, L539, G559, S564, V566, S569, Q570, and

F573 Interestingly, of the two residues in this pocket that contribute to selectivity for cAMP over cGMP

in HsPDE4, only Q570 (corresponding to Q443 in HsPDE4B2) is conserved, while the residue corres-ponding to N395 in HsPDE4B2 is substituted by A524 The side chain of this N395 in the structure of HsPDE4B forms two hydrogen bonds with adenine, with the 6-NH2 atom and with the 7-N ring atom, while it might only form a single interaction with either the 7-N atom or the 2-NH2 atom of guanine While the presence of an N residue at this position favors cAMP binding, it does not preclude cGMP binding However, in several of the human PDEs that accept cGMP as a substrate, the position of N395 is substituted by an alanine (HsPDE5 and HsPDE6) or glycine (HsPDE3 [27]) The presence of an alanine resi-due at this position implies that TcrPDEC might be capable of using cGMP as a substrate This could be experimentally confirmed (see below) Compared with the set of 19 residues that are completely conserved

in all 11 human PDEs [27], four substitutions in

Fig 3 Dimeric structure of the FYVE-type domain (A) Gel filtration

(Superdex 75 PC 3.2 ⁄ 30) of an N-terminal fragment of TcrPDEC

containing the FYVE-variant domain through the coiled-coil region

(amino acids 1–272) (B) Gel filtration of FYVE-type domain alone

(amino acids 1–74 of TcrPDEC) Elution positions of the

FYVE-con-taining polypeptides are indicated by asterisks Vertical black arrow:

elution position of TEF protease (27 kDa) Elution position of

molecular weight markers (open arrows): 1, aldolase (158 kDa); 2,

bovine serum albumin (67 kDa); 3, chymotrypsingen A (25.0 kDa);

4, aprotinin (6.5 kDa); 5, vitamin B12 (1.35 kDa).

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TcrPDEC are notable In the predicted helix 9, a

con-served alanine is substituted by S429 in TcrPDEC In

predicted helix 10, the first of the two vicinal histidines

is replaced by L441, and in predicted helix 11, a

con-served alanine is substituted by H479 Finally, in

pre-dicted helix 14, a conserved aspartate is replaced by

E547 The three substitutions represented by L441, H479

and E547 are specific for TcrPDEC Another class1

PDE of T cruzi, TcPDE1, that has a low Km and is

cAMP-selective [14], conforms to the mammalian PDE

pattern in all three positions These three substitutions

in TcrPDEC may contribute to its dual-substrate

spe-cificity, and⁄ or to its relatively high Km for both

sub-strates (see below)

Functional complementation of a PDE-deficient

S cerevisiae strain Deletion of the two PDE genes ScPDE1 and ScPDE2 from the S cerevisiae genome leads to an accumula-tion of cAMP in the cells, leading to marked heat-shock sensitivity [29] Heterologous complementation

of the heat-shock sensitivity phenotype of PDE-defici-ent yeast strains has proven to be a highly sensitive functional validation for suspected PDE genes [20–32] The full-size open reading frame of TcrPDEC, as well

as the predicted catalytic domain (T291–S657) were expressed in the PDE-deficient S cerevisiae strain PP5 [31] In addition, the same two constructs carrying

Fig 4 Sequence alignment of catalytic domains Grey vertical boxes indicate residues that are identical in all human PDEs as well as in TcPDE1 and TcrPDEC; open vertical boxes indicate residues that are conserved in all 11 human PDEs and in TcPDE1, but are substituted in TcrPDEC; black dots represent residues that are necessary for positioning the catalytically important histidine residue (arrow); boxes indicate the consensus helices; asterisk and bold, asparagine residue which confers cAMP preference (N395in HcPDE4B) m, metal binding pocket;

q, Q-domain [28].

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N-terminal hemagglutinin tags were also expressed.

Transformants were patched and tested for heat shock

resistance (Fig 5) All four constructs fully restored

the heat shock resistance phenotype to the indicator

strain The results of these complementation

experi-ments established that TcrPDEC codes for a functional PDE, and that this enzyme can use cAMP as a sub-strate

Characterization of catalytic activities Soluble cell lysates were prepared from yeast strains expressing either full-length TcrPDEC or its catalytic domain only The recombinant PDE activity was entirely dependent on the presence of divalent cations, with Mg2+ providing the best activity Setting the activity observed with 2 mm MgCl2 as 100%, 2 mm MnCl2 yielded 53.5 ± 15.9% activity, 0.2 mm CaCl2

15.3 ± 1.9%, and 30 lm ZnCl2 19.5 ± 3.2% The activity obtained with 2 mm MgCl2 was slightly, but consistently, further stimulated by the presence of 0.2 mm CaCl2 (116.1 ± 13.2%) In all subsequent experiments, reactions contained 10 mm MgCl2 Michaelis-Menten kinetics were determined with cAMP or cGMP as substrates (Fig 6A,B) TcrPDEC exhibits a Kmfor cAMP of 31.6 ± 9 lm (N¼ 6) This

is significantly higher than the Kmfor cAMP observed for a previously characterized PDE from T cruzi, TcPDE1 [14] Unexpectedly, TcrPDEC also hydrolyzes cGMP with a similar Vmax, and with a Km of 78.2 ± 25 lm (n¼ 3) This dual substrate specificity might reflect the presence of the A524 residue in the sequence of TcrPDEC, replacing an asparagine residue

- heat shock + heat shock

pLT-TcPDEC

pLT-TcPDEC-cat pHA-TcPDEC

pHA-TcPDEC-cat pLT1 pd6

Fig 5 Reversal of the heat-shock sensitivity phenotype of S

cere-visiae PDE deletion strain PP5 Duplicate patches of independent

transformants of S cerevisiae PP5 incubated with (left) or without

(right) an initial heat shock at 55 C for 15 min pLT-TcrPDEC,

full-length TcrPDEC; pHA-TcrPDEC, full-full-length TcrPDEC with a

N-ter-minal hemagglutinin tag; pLT-TcrPDEC-cat, catalytic domain of

TcrPDEC (W 367 -H 597 ); pHA-TcrPDEC-cat, catalytic domain of

TcrP-DEC with an N-terminal hemagglutinin tag; pLT1, empty vector

(negative control); pd6, TbPDE1 (positive control).

Dipyridamole

Etazolate

[Inhibitor] (µM) [Inhibitor] (µM)

Trequinsin

IBMX

µM cAMP

mo

µM cGMP

MP

D

C

Fig 6 Michaelis-Menten plots (A) with

cAMP as substrate and (B) with cGMP as

substrate (C) and (D) show representative

plots of IC 50 determinations for various PDE

inhibitors (substrate concentration: 1 l M

cAMP).

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that appears to confer cAMP selectivity in the

cAMP-specific PDEs This dual cAMP-specificity is a novel feature

for a kinetoplastid PDE since all PDEs characterized

in these organisms so far are entirely cAMP selective

[13,15,16,30,32, Johner et al submitted]

Inhibitor profiling

To explore the sensitivity of TcrPDEC against a

spec-trum of inhibitors, the potency of a number of

com-mercially available PDE inhibitors was determined

using 1 lm cAMP as the substrate Most of the

com-pounds tested exhibited low potency against TcrPDEC

(Table 1), though several of them are high-potency

inhibitors of various human PDEs Only three of the

compounds tested, trequinsin, etazolate, and

dipyrida-mole, exhibited IC50 values below 10 lm (Figs 6C.D)

With human PDEs, these three compounds inhibit

different PDE families with high potency (trequisin:

HsPDE3, IC50 0.0003 lm; etazolate: HsPDE4, IC50

0.55 lm; dipyridamole: HsPDE5, IC50 0.9 lm) All

three compounds have also proven to be effective

inhibitors of various PDE families from different

kine-toplastids (15,16,30,32, Johner et al submitted] The

IC50 value of the broad-spectrum PDE inhibitor

iso-butyl methyl xanthine (IBMX) against TcrPDEC is

68 lm (Fig 6D) This potency is similar to that found

for IBMX with many of the human PDEs However,

it is considerably higher than what was found for other

trypanosomatid PDEs such as TcPDE1 [14], TbPDE1 [30] or TbPDE2 [16,32]

Discussion TcrPDEC represents a novel type of class I PDE, as defined by the sequence characteristics of its catalytic domain [17] All PDEs of this class exhibit a similar overall architecture, where the N-terminal moiety con-tains various assortments of regulatory domains and the C-terminal part hosts the catalytic domain Tcr-PDEC is a notable exception to this rule, in that its catalytic domain is localized in the middle of the poly-peptide chain (amino acids T291–S657of a total of 924) The C-terminal portion contains no recognizable func-tional domains or motifs In the amino acid sequence

of the catalytic domain, all residues that have been identified as important for substrate recognition, selec-tivity and catalysis [27,28] are fully conserved In the structure of human PDE4, two residues within the hydrophobic purine-binding pocket were shown to be crucial for purine binding and for adenine-specificity (in HsPDE4B2: Q443and N395, respectively) The pres-ence of an asparagine in this position favors the bind-ing of cAMP, but does necessarily preclude the binding of cGMP However, in several of the human PDEs that accept cGMP as their substrate, the posi-tion of N395is substituted by alanine (in HsPDE5 and HsPDE6) or by glycine (in HsPDE3) [27]

Interest-Table 1 Potency of selected PDE inhibitors All IC50values were determined at 1 l M substrate.

Inhibitor

IC50(l M )

Mammalian PDE selectivity (IC 50 l M )

PDE 6 (0.4) PDE 8 (4.5) PDE 10 (1.1) PDE 11 (1–2)

PDE 4 (0.2–0.8)

*n.s., nonselective inhibitor; † isobutyl methyl xanthine; ‡ 8-methoxymethyl-IBMX; EHNA, erythro-9-(2-hydroxy-3-nonyl)adenosin.

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ingly, the position corresponding to N395 in

HsPDE4B2 is occupied by an alanine residue (A524) in

TcrPDEC Based on the information available from

the human PDEs, this finding predicts that TcrPDEC

could accept cGMP as a substrate This prediction

could be experimentally verified Recombinant

TcrP-DEC proved to be a dual-specificity PDE, with Km

values of 32 lm for cAMP and 78 lm for cGMP,

respectively These Km values are relatively high when

compared with most of the human and trypanosomal

PDEs Nevertheless, they are similar to those of the

human dual-specificity PDE HsPDE2 (30–50 lm for

cAMP and 15–30 lm for cGMP [33])

While the observation that TcrPDEC can hydrolyze

cGMP is in perfect agreement with the prediction from

its amino acid sequence, the biological significance of

this finding is not at all clear Paveto et al have

pre-sented evidence that T cruzi might contain a cGMP

signaling pathway that can be stimulated by nitric

oxide [4] In other kinetoplastids such as L major or

T brucei, no cGMP PDE activity was detectable in

whole cell lysates (unpublished data) Database

screen-ing of kinetoplastid genomes has not so far identified

any genes for putative soluble guanylyl cyclases, as

would have been predicted by the work of Paveto

et al [4] However, one cannot exclude that one or

several of the predicted adenylyl cyclases identified in

the T cruzi and other kinetoplastid genomes might

actually function as a guanylyl cyclase In mammalian

adenylyl and guanylyl cyclases, a single amino acid

substitution can determine the substrate specificity [34]

A second unusual feature of TcrPDEC is the

pres-ence of a FYVE-type domain at its N-terminus FYVE

domains have been identified in numerous proteins,

but so far never in a PDE While some FYVE

domains of some proteins were shown to interact with

PtdIns(3)P and to locate to endosomal membranes, the

functional role of the FYVE-variant and

FYVE-rela-ted domains is less clear The FYVE-type domain of

TcrPDEC is followed by two closely spaced coiled-coil

regions Such coiled-coil domains were shown to serve

to dimerize the FYVE domains in EEA1 [26] In

con-trast, gel filtration analysis of the FYVE-type domain

of TcrPDEC has now shown that it can form a stable

dimer in the absence of the coiled-coil domains This

dimerization might enhance the interaction of the

FYVE-type domain with a target membrane On the

other hand, the dimerization will also entail a

dimeri-zation of the downstream catalytic domains, and may

thus influence their catalytic properties The coiled-coil

region of TcrPDEC may mediate an additional level

of organization, the function of which remains to be

explored The overall concept of dimerization and

possibly modulation of the activity of PDE catalytic domains via their regulatory N-terminal regions has been established for human HsPDE2 and HsPDE5, where dimerization takes place via GAF domains [35]

A similar GAF-mediated dimerization and modulation also occurs in TbPDE2B of T brucei [36] and possibly also in TbPDE2C

The overall sequence of the FYVE-type domain of TcrPDEC exhibits a smaller overall electrical charge than the canonical FYVE domains The functional consequences of this are not clear, as we have not been able to demonstrate binding of a recombinant FYVE-type domain of TcrPDEC to individual phospholipids

in a spot assay While this naive approach works with certain FYVE domains [24,25], it is certainly less than definitive, since the binding requirements for individual FYVE domains may be more complex than just indi-vidual phospholipids spotted on a solid surface If the FYVE-type domain of TcrPDEC does in fact interact with membranes, it may serve to confer a precise sub-cellular localization to the enzyme This is a most interesting possibility, as the role of subcellular local-ization assumes an ever greater significance in the emerging concepts of intracellular signal transduction [37] In fact, being in the right place at the right time may be the preeminent requirement for the proper working of a signaling enzyme such as a PDE

Experimental procedures Materials

Radiochemicals were purchased from Moravek Biochemi-cals Inc (Hartmann Analytik, Zurich, Switzerland) cAMP and cGMP (sodium salts) were obtained from Sigma (Buchs, Switzerland) PDE inhibitors were from the follow-ing sources: I I IBMX, papaverine, milrinone, trequin-sin, 8-methoxymethyl-IBMX, dipyridamole and rolipram were from Sigma; erythro-9-(2-hydroxy-3-nonyl)adenosin (EHNA), zardaverine, cilostamide and Ro-20–1724 were from BioMol (Anawa Trading, Wangen, Switzerland); etaz-olate and pentoxifylline were from Calbiochem (Juro Sup-plies, Lucerne, Switzerland) DNA sequencing reactions were run with BigDye terminators (PE-Biosystems) and were analyzed on an ABI Prism 377 instrument Genomic DNA was extracted from the T cruzi promastigotes sup-plied by R Brun, Swiss Tropical Institute, University of Basel

Identification and cloning of TcPDE-C

A search of the T cruzi database (http://www.genedb.org/ genedb/tcruzi) for PDE-coding sequences identified an open

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reading frame (Tc00.1047053506697.20) that appeared to

code for a highly unusual PDE, termed TcrPDEC Its

entire coding region was amplified by high-fidelity PCR

(Expand High Fidelity PCR System, Roche Diagnostics,

Rotkreuz, Switzerland) using T cruzi genomic DNA as the

template The primers used were TcPDE4 for (5¢-CAG

TCGACATATGTCGGAGGACGCTGGGCTTC-3¢) and

TcPDE4-rev (5¢-CGTGGATCCTCAGCACTGCGTCAA

CAGAGTG-3¢) The PCR product was cloned into the

pCR2.1-TOPO vector The predicted catalytic region of

TcrPDEC (T291–S657) was amplified separately, using

prim-ers TcPDE4-catf (5¢-CAGTCGACATATGACAATACT

CGCAGTTGTTCC-3¢) and TcPDE4-catr (5¢-CTGGAT

CCTCAACTGGCTGTTCTCAGCTCCTG-3¢) and cloned

into pGEM-T Easy plasmid vector (Promega, Catalys,

Wallisellen, Switzerland) All cloned PCR products were

verified by DNA sequencing

Expression in S cerevisiae

For expression in S cerevisiae, the entire open reading

frame and the catalytic region (T291–S657) of TcrPDEC were

cloned via SalI and BamHI restriction sites into two variants

of the yeast expression vector pLT1 [30] One variant directs

the expression of the genuine protein, while the other adds

an N-terminal hemagglutinin-tag to facilitate detection of

the recombinant proteins Transformation of the constructs

into the PDE-deficient S cerevisiae strain PP5 (MATa leu2–

3 leu2–112 ura3–52 his3–532 his4 cam pde1::ura3 pde2::

HIS3) was carried out as described earlier [30,31]

Complementation assay

The heat-shock assay to detect complementation of the

PDE-deficient phenotype of the S cerevisiae strain PP5 was

carried out exactly as described [30] Single yeast colonies

were replica patched onto YPD plates (10 gÆL Bacto-Yeast

extract, 20 gÆL Bacto-Peptone, 20 gÆL glucose, 20 gÆL

bacto-agar) prewarmed to 55C, and were incubated for another

15 min at 55C Plates were then cooled to room

tempera-ture and incubation was continued at 30C for 18–36 h

Yeast cell lysis

Yeast cell lysis was performed as described [32] with minor

modifications Briefly, yeast cells grown to mid-log to

end-log phase in SC-leu medium were collected, resuspended in

the original volume of prewarmed YPD medium, and

incu-bated for an additional 3.5 h at 30C to maximize protein

expression Cells were washed twice in H2O, and the washed

cell pellet was stored at)70 C The pellet was thawed on

ice and suspended in an equal volume of ice-cold extraction

buffer [50 mm Hepes pH 7.5, 100 mm NaCl, 1· Complete

protease inhibitor cocktail with EDTA (Roche

Diagnos-tics)] Cells were lysed by grinding with glass beads (0.45– 0.50 mm) in 2 mL Sarstedt tubes, using a FastPrep FP120 cell disruptor (3· 45 s at setting 4) The subsequent centrif-ugation steps were carried out exactly as described Glycerol was added to the resulting supernatant to a final concentra-tion of 15% (v⁄ v), aliquots were snap-frozen in liquid nitro-gen and were stored at )70 C Protein expression and stability of the enzyme under assay conditions were monit-ored by immunoblotting, using a monoclonal antibody against the hemagglutinin tag (Roche Diagnostics)

Protein–lipid overlay assay Protein–lipid binding studies were performed as described [38], with the following modifications: Lipids were dissolved

in chloroform⁄ methanol ⁄ water (1 : 2 : 0.8 by volume) A 20–2500-pmol amount of each lipid were spotted onto nitrocellulose filters and were air-dried for 1 h Membranes were blocked with 3% (w⁄ v) fat-free bovine serum albumin (Sigma) in 10 mm Tris⁄ HCl, pH 8.0, 150 mm NaCl, 0.1% (v⁄ v) Tween-20) for 1.5 h at room temperature Filters were incubated with 2 lgÆmL)1 of recombinant GST-fusion pro-teins overnight at 4C After washing the filters five times with incubation buffer, the bound GST fusion protein was quantitated by developing the filters with GST body, followed by horse radish conjugated IgG anti-body

Phosphodiesterase assay PDE activity was determined by a modification of the two-step procedure of Thompson and Appleman [39] as des-cribed previously (Johner et al submitted) PDE activity was determined in 50 mm Hepes pH 7.5, 0.5 mm EDTA,

10 mm MgCl2, 50 lgÆmL)1bovine serum albumin in a final volume of 100 lL Each assay contained 50 000 cpm

3

H-labelled cAMP or cGMP, with unlabeled cAMP or cGMP added to the desired total substrate concentration Reactions were run at 30C for 15 min They were then stopped by the addition of 25 lL of 0.5 m HCl, neutralized with 20 lL 1 m Tris base, and digested with 10 lL of calf intestinal alkaline phosphatase (Roche Diagnostics; 1 unit per 10 lL) for 15 min at 37C After dephosphorylation, the reactions were applied to 1 mL columns of QAE-Sepha-dex A25 in 30 mm ammonium formate, pH 6.0 The

3H-adenosine or 3H-guanosine formed during the reaction was eluted with 1.6 mL of 30 mm ammonium formate,

pH 6.0 into 3.5 mL water-miscible scintillation fluid (Pack-ard Ultima Flo; Pack(Pack-ard Bioscience, Zurich, Switzerland)

In all reactions no more than 20% of the substrate was hydrolyzed Assays were always carried out in triplicates, and at least three independent experiments were performed

IC50studies were all carried out at 1 lm substrate concen-tration Inhibitors were dissolved in dimethyl sulfoxide

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The dimethyl sulfoxide (DMSO) concentration in the

final assay solutions never exceeded 1%, and appropriate

control reactions with DMSO alone were always included

Data were analyzed using the GraphPad Prism software

package

Gel filtration analysis of the FYVE-variant domain

PCR-amplified DNA fragments corresponding to amino

acids 1–74 of TcrPDEC (FYVE-variant domain alone) and

1–272 (FYVE-variant domain plus coiled-coil region) were

cloned into the expression vector pGST-parallel2 [40]

The recombinant proteins are N-terminally fused to

glutathione-S-transferase, from which they are separated by

a TEV protease cleavage site Recombinant protein from

125 mL bacterial culture were purified by batch-adsorption

to 125 lL Glutathione-Uniflow resin (BD Biosciences,

Basel, Switzerland) for 2 h at 4C The resin was washed

four times with ice-cold 50 mm Tris⁄ HCl, pH 7.5, 150 mm

NaCl, 10 mm 2-mercaptoethanol, 5 mm sodium citrate,

10 lm ZnCl2 and 5% glycerol After washing, the beads

were suspended in an equal volume of washing buffer,

30–50 units of AcTEV protease (Invitrogen; Juro Supplies,

Lucerne, Switzerland) were added, and the slurry was

incu-bated for 36 h at 6C on a rotating wheel The beads were

then pelleted by centrifugation, and the supernatant

con-taining the released FYVE-variant domain was purified by

centrifugation through a 0.2 lm filter

Proteins were analyzed on a Superdex 75 PC 3.2⁄ 30 gel

filtration column, using the Pharmacia Smart system The

column was precalibrated with the following markers:

aldo-lase (158 kDa), bovine serum albumin (67 kDa), ovalbumin

(44 kDa), chymotrypsinogen A (25 kDa), RNase A

(13.7 kDa), aprotinin (6.5 kDa) and vitamin B12 (1.35 kDa)

All fractions were subsequently analyzed by gel

electrophor-esis The larger FYVE-variant⁄ coiled-coil polypeptide was

further analyzed on a Superdex 200 PC 3.2⁄ 30 column

calib-rated with the same markers, plus the following additional

ones: thyroglobulin (670 kDa), ferritin (450 kDa) and

cata-lyze (232 kDa)

Acknowledgements

We would like to thank M Linder and X Lan Vu for

their technical assistance, R Brun of the Swiss

Trop-ical Institute, University of Basel, for T cruzi cultures,

the laboratory of J H Hurley (NIH, Bethesda, MD,

USA) for providing expression plasmids for

Vps27p-FYVE-GST fusion proteins, Peter Buetikofer (Institute

of Molecular Biology, University of Bern) for

provi-ding phospholipids and valuable advice This work

was supported by grant Nr 3100–067225 of the Swiss

National Science Foundation, and by the COST B22

programme of the European Union

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