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Perham Cambridge Centre for Molecular Recognition, Department of Biochemistry, University of Cambridge, UK Limited proteolysis of the pyruvate decarboxylase E1, a2b2 component of the pyr

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Site-directed mutagenesis of a loop at the active site of E1 (a2b2)

of the pyruvate dehydrogenase complex

A possible common sequence motif

Markus Fries*, Hitesh J Chauhan†, Gonzalo J Domingo‡, Hyo-Il Jung§and Richard N Perham

Cambridge Centre for Molecular Recognition, Department of Biochemistry, University of Cambridge, UK

Limited proteolysis of the pyruvate decarboxylase (E1, a2b2)

component of the pyruvate dehydrogenase (PDH)

multi-enzyme complex of Bacillus stearothermophilus has indicated

the importance for catalysis of a site (Tyr281-Arg282) in the

E1a subunit (Chauhan, H.J., Domingo, G.J., Jung, H.-I &

Perham, R.N (2000) Eur J Biochem 267, 7158–7169) This

site appears to be conserved in the a-subunit of

hetero-tetrameric E1s and multiple sequence alignments suggest

that there are additional conserved amino-acid residues in

this region, part of a common pattern with the consensus

sequence -YR-H-D-YR-DE- This region lies about 50

amino acids on the C-terminal side of a 30-residue motif

previously recognized as involved in binding thiamin

diphosphate (ThDP) in all ThDP-dependent enzymes The

role of individual residues in this set of conserved amino acids in the E1a chain was investigated by means of site-directed mutagenesis We propose that particular residues are involved in: (a) binding the 2-oxo acid substrate, (b) decarboxylation of the 2-oxo acid and reductive acety-lation of the tethered lipoyl domain in the PDH complex, (c) an open–close mechanism of the active site, and (d) phosphorylation by the E1-specific kinase (in eukaryotic PDH and branched chain 2-oxo acid dehydrogenase com-plexes)

Keywords: pyruvate dehydrogenase; multienzyme complex; thiamin diphosphate; limited proteolysis; enzyme mecha-nism

The family of 2-oxo acid dehydrogenase (2-OADH)

multi-enzyme complexes contains three members: the pyruvate

dehydrogenase (PDH), the 2-oxoglutarate dehydrogenase

(OGDH) and the branched-chain 2-oxo acid dehydrogenase

(BCDH) complexes These are responsible for the oxidative decarboxylation of pyruvate, 2-oxoglutarate and branched-chain 2-oxo acids, respectively, in each case generating the corresponding acyl-CoA and NADH The complexes all occupy important positions in the metabolism of the cell and generally comprise three component enzymes: a 2-oxo acid decarboxylase (E1, EC 1.2.4), a dihydrolipoyl acyl-transferase (E2, EC 2.3.1), and dihydrolipoyl dehydroge-nase (E3, EC 1.8.1.4) E1 catalyses the first and irreversible step of the overall reaction, the thiamin-diphosphate (ThDP)-dependent oxidative decarboxylation of the 2-oxo acid, followed by the reductive acylation of a lipoyl prosthetic group covalently bound to a lysine residue in the lipoyl domain of the E2 chain The reaction catalysed by E1 is rate-limiting for the overall activity of the complex [1,2], probably at the reductive acylation step [2,3] The E2 component catalyses the transfer of the acyl group from the lipoyl-lysine group to CoA, and the cycle is completed by reoxidation of the resulting dihydrolipoyl group catalysed

by E3, a flavoprotein, generating NADH and H+from NAD+ For recent reviews, see de Kok et al and Perham [4,5]

In eukaryotic PDH and BCDH complexes, modulation

of catalytic activity is achieved by phosphorylation-dephosphorylation of E1; an E1-specific kinase inactivates E1 by phosphorylating certain serine residues in the E1a component and activity is restored by the action of a specific phosphatase [6–8] Depending on the organism and the type

of 2-OADH complex, the E1 component exists either as a heterotetramer (a2b2) or a homodimer (a2) In the PDH complexes of Gram-negative bacteria and in all OGDH complexes, E1 is a homodimer; in PDH complexes of

Correspondence to R N Perham, Department of Biochemistry,

University of Cambridge, Sanger Building, Old Addenbrooke’s Site,

80 Tennis Court Road, Cambridge CB2 1GA, UK.

Fax: + 44 1223 333667, Tel.: + 44 1223 333663,

E-mail: r.n.perham@bioc.cam.ac.uk

Abbreviations: BCDH, branched-chain 2-oxo acid dehydrogenase;

DCPIP, 2,6-dichlorophenolindophenol; E1, 2-oxo acid decarboxylase;

E1a, alpha subunit of E1; E1b, beta subunit of E1; E1p, pyruvate

decarboxylase of PDH complex; E2p, dihydrolipoyl acetyltransferase;

E3, dihydrolipoyl dehydrogenase; IPTG, isopropyl thio-b- D

-galacto-side; OGDH, 2-oxoglutarate dehydrogenase; PSBD, peripheral

subunit-binding domain; SPR, surface plasmon resonance;

ThDP, thiamin diphosphate.

Enzymes: pyruvate decarboxylase (EC 1.2.4.1); dihydrolipoyl

acetyl-transferase (EC 2.3.1.12); dihydrolipoyl dehydrogenase (EC 1.8.1.4).

*Present address: Cambridge Institute for Medical Research,

Wellcome Trust/MRC Building, Box139 Addenbrooke’s Hospital,

Hills Road, Cambridge CB2 2XY, UK.

Present address: Adprotech Ltd, Chesterford Research Park, Little

Chesterford, Saffron Walden, Essex CB10 1XL, UK.

Present address: Seattle Biomedical Research Institute, 4 Nickerson

Street, Seattle, WA 98109, USA.

§Present address: Wolfson Institute for Biomedical Research, The

Cruciform Building, Gower Street, London WC1E 6BT, UK.

(Received 19 September 2002, revised 5 December 2002,

accepted 20 December 2002)

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Gram-positive bacteria and eukaryotes and in BCDH

complexes, E1 is a heterotetramer [9] In all cases, E1 acts as

a functional dimer with two active sites of equal catalytic

efficiency [10,11] and two ThDP per E1, albeit bound with

different affinities [11–13] There is some evidence that the

two active sites of pigeon breast muscle E1p work in an

alternating site mechanism and that intersubunit

inter-actions may play an essential part in the catalysis by, and

regulation of, the enzyme [11] This is consistent with the

finding that phosphorylation of only one of two active sites

in mammalian E1p leads to complete inactivation [7]

Crystal structures of the heterotetrameric E1s from the

Pseudomonas putida[14] and human [15] BCDH complexes,

both E1(a2b2) heterotetramers, are available (Fig 1) The

E1a chain houses a conserved ThDP-binding site [16], the

phosphate groups of ThDP being anchored by residues of

this sequence motif through a bound Mg2+ion, and the

aminopyrimidine end bound by the b-subunit [17] Thus,

both subunits are needed for a catalytically active enzyme

Limited proteolysis of E1 (a2b2) from the PDH

com-plexes of Bacillus subtilis [18] and pig heart [19] leads to

cleavage of the E1a subunit, whereas the E1b subunit

remains intact A detailed study of the limited proteolysis of

the E1 component of B stearothermophilus PDH complex [20] identified Tyr281 (for chymotrypsin) and Arg282 (for trypsin) of the E1a subunit, two conserved residues in a loop

at the entrance to the active site (Fig 1), as the main cleavage sites Cleavage of the E1a-subunit activated the enzyme, as determined by a decarboxylation assay of the free E1 in the presence of an artificial electron acceptor, but the catalytic activity of a reconstructed PDH complex was found to be substantially lowered Multiple sequence alignments reveal additional conserved residues in the region of Tyr281 and Arg282 We describe here a study of the role of these conserved residues and compare the results with those of limited proteolysis in ascribing possible functions to them

Materials and methods Materials

Restriction endonucleases were obtained from Pharmacia Biotech (St Albans, UK) or New England Biolabs (Hitchin, UK), Pfu DNA polymerase was from Stratagene (Cam-bridge, UK) and T4 DNA ligase from Promega

Fig 1 Crystal structure of heterotetrameric (a 2 b 2 ) E1 (A) P putida E1b [14] (B) Human E1b [15] The two a-subunits are shown in orange and red, the two b-subunits in light blue and dark blue The cofactor

ThDP-Mg2+, located at the interface between an a-and b-subunit, is indicated in spacefill Residues 301–314 are not included in the human E1b structure, so that not all residues

of the sequence motif could be highlighted (C) Conserved active site motif derived from the crystal structure of P putida E1b (D) Conserved active site motif derived from the crystal structure of human E1b.

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(Southampton, UK) Isopropyl thio-b-D-galactoside

(IPTG) and phenylmethanesulfonyl fluoride were purchased

from Melford Laboratories (Chelsworth, UK),

bacterio-logical media from Beta Laboratory (West Molesely, UK)

and Duchefa (Haarlem, the Netherlands), and ampicillin

from Beecham Research Laboratories (Brentford, UK)

Pyruvate, NAD+, ThDP, 2,6-dichlorophenolindophenol

(DCPIP), coenzyme A and FAD were from Sigma (Poole,

UK) Solid [2-14C]pyruvic acid, sodium salt (specific activity,

15.9 mCiÆmmol)1) was from New England Nuclear

(Bos-ton, MA, USA) Centrifugal filter devices were purchased

from Millipore

Bacterial strains and plasmids

E coli strain TG1recO and plasmids pKBstE1a and

pKBstE1b, expressing genes encoding the B

stearother-mophilus E1a and E1b subunits, respectively, have been

described previously [21] E coli strain BL21 (DE3) [B,

F–, ompT, hsdSB(rB- , mB-), gal, dcm (DE3)] from Novagen

(Madison, USA) was employed to express the B

stearo-thermophilusE3 gene (from pBSTNAV/E3) and E2p gene

(from pETBstE2) [21] and a subgene encoding a di-domain

that comprises the lipoyl domain, the peripheral

subunit-binding domain and the linker between them (from

pET11d2D and pET11ThDD) The subgene in plasmid

pET11d2D encodes residues 1–171 of the wild-type B

ste-arothermophilus E2p chain [22]; plasmid pET11ThDD

encodes the same di-domain but with a thrombin-cleavage

site inserted in the linker region between the lipoyl and

binding domains [23] Lipoate protein ligase A was purified

from E coli BL21 (DE3) cells transformed with the plasmid

pTM202 [24]

Recombinant DNA techniques and mutagenesis

Recombinant DNA techniques were carried out as described

elsewhere [25] DNA fragments were isolated from gels using

the QIAquick gel extraction kit, and plasmids were prepared

by means of the Qiagen plasmid kit, both from Qiagen

(Hilden, Germany) Site-specific mutations were introduced

into the plasmid pKBstE1a using splicing-by-overlap

exten-sion PCR [26] The fidelity of the amplified DNA fragments

was established by DNA sequence analysis after subcloning

into the vector All the sequences differed slightly from the

original pKBstE1a plasmid [21] in the noncoding region

following the stop codon The differences (four C instead of

three C at positions 1405–1407, and two additional G after

position 1435) were the same in all instances, suggesting that

there may have been errors in this part of the original

pKBstE1a sequence

Protein purification

Purification of wild-type and mutant E1s was carried out

as described previously [21,27] The E1aS283C mutant

was purified with 1 mM dithiothreitol added to all

buffers to prevent formation of disulfide bonds E2

and E3 were purified as described elsewhere [28] The

E2p domain [29] and the thrombin-cleavable

di-domain and the lipoyl di-domain subsequently released by

thrombin treatment [23] were purified essentially as

described elsewhere The apo-forms of the lipoyl domain, di-domain and the E2 component were lipoylated by means of lipoate protein ligase A, also essentially as described elsewhere [22,30]

Enzyme assays The E1 component was assayed for catalytic activity by means of three different assays

DCPIP assay This measures the rate of reduction of the artificial electron acceptor, DCPIP, by the E1 component [31] The decrease in A600was monitored at a temperature of

30C The assay mixture contained 0.2 mMThDP, 2 mM

MgCl2, 50 lM DCPIP, 100 mM potassium phosphate,

pH 7.0 and 20 lg of E1 The reaction was started by adding pyruvate (final concentration 400 lM) after the assay mix had been incubated for 10 min at 30C

Reductive acetylation assay This measures the reductive acetylation of the free lipoyl domain with [2-14C]pyruvate at

25C [30,32] The assay mixture (350 lL) contained 0.2 mM ThDP, 1 mM MgCl2, 0.3 mgÆmL)1 BSA, 20 mM

potassium phosphate, pH 7.0, plus 3 nmol lipoyl domain and 5 lg E1 The reaction was started by addition of14 C-labelled pyruvate (1 lCi, about 60 nmol) after the assay mix had been incubated for 10 min at 25C

PDH assay This measures the rate of formation of NADH at 340 nm and 30C after the complex has been reconstituted from its individual components [1,33,34] The assay contained 0.2 mM ThDP, 1 mM MgCl2, 2.6 mM

cysteine HCl, 2 mM pyruvate, 0.13 mM CoA, 50 mM

potassium phosphate, pH 7.0 E1, E2 and E3 were added

in molar ratios of E1(a2b2)/E2(a)/E3(a2) of 3 : 1 : 3; the amount of E2 used was 0.5 lg, 1.0 lg, 1.5 lg and 2.0 lg The reaction was started by adding the pyruvate and CoA after the PDH complex had been allowed to assemble in the assay mixture for at least 3 min at 30C Specific activities for the reconstituted PDH complex are expressed

as U per mg E2 All assays were normally carried out

at least three times and the results are expressed as the mean ± SD

Determination of kinetic parameters

Kmvalues for pyruvate and ThDP were determined using the DCPIP assay by varying the concentrations of pyruvate from 0.2 lMto 4000 lMand the ThDP concentrations from 0.04 lMto 200 lM When determining the kinetic param-eters for ThDP, the pyruvate concentration was kept at

1 mM Data were analysed and fitted to a Michaelis– Menten curve using theSIGMA PLOTsoftware

Temperature-dependence of catalytic activity The dependence of the catalytic activity of E1 on tempera-ture was investigated using the DCPIP assay over a temperature range of 15C to 85 C Assay mixtures were incubated for exactly 10 min at the relevant temperature before the reaction was started by the addition of pyruvate and then monitoring the decrease in A

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Interaction of E1 with the peripheral subunit-binding

domain

Mixtures of E1 and di-domain were submitted to

non-denaturing PAGE using the Pharmacia Phast System The

interaction of di-domain with E1 was also investigated using

surface plasmon resonance detection (BIAcore, Pharmacia

Biosensor AB), immobilizing the lipoyl domain by means of

its lipoyl group Both sorts of experiment were carried out as

described in detail elsewhere [35]

Results

Multiple sequence alignment of E1 components

of 2-OADH complexes

No obvious sequence homology can be detected between

homodimeric forms of E1p and E1o Likewise, amino-acid

sequences of heterotetrameric (a2b2) E1s do not readily

align with sequences of homodimeric E1s However, a

sequence motif of about 30 amino acids, beginning with a

highly conserved amino-acid triplet of GDG and ending

with NN, has been identified as common to all

ThDP-dependent enzymes [16], and confirmed as a ThDP-binding

motif in the crystal structures of several ThDP-utilizing

enzymes [17] Aside from this motif, no active site residues

other than those directly in contact with ThDP have been

reported as conserved [36]

However, inspection of the available E1 sequences

suggests that there may be another set of amino-acid

residues common to the E1 (a2b2) components of 2-OADH

complexes This consensus set,

-Y/F/WR-H-D-YR-D/EE-in the E1a cha-Y/F/WR-H-D-YR-D/EE-ins (Fig 2), -Y/F/WR-H-D-YR-D/EE-includes the Y281R282 site of

limited proteolysis [20] and lies about 50 amino-acid

residues to the C-terminal side of the ThDP-binding motif

The crystal structures of P putida [14] and human [15] E1b

show them as located on a loop region in Ela close in space

to the cofactor ThDP-Mg2+(Fig 1) Also included in this

region are two serine residues in the Ela chain of

mamma-lian PDH and BCDH complexes that serve as

phosphory-lation sites 1 and 2, responsible for inhibiting E1 when they

become phosphorylated Other consistently conserved

resi-dues in the aligned sequences appear to mark the beginning

and end of the loop region: notably -PXXXE- (where X is

generally a large aliphatic residue) at the N-terminal end,

and -DP- (or -DHP-) at the C-terminal end (Fig 2)

Choice of mutations

The importance of the loop region in the a-subunit of the

B stearothermophilusE1p component was highlighted by its

susceptibility to limited proteolysis with trypsin and

chymo-trypsin and the effects of the cleavages on catalytic activity

[20] In the present study, residues Phe266, Arg267 and

Asp276 in the loop region of B stearothermophilus E1a were

replaced with alanine, as were Tyr281 and Arg282, the sites of

cleavage with chymotrypsin and trypsin, respectively

Like-wise the neighbouring residue, Ser283, corresponding to a

phosphorylation site in eukaryotic BCDH complexes, was

also replaced with alanine The Y281S/R282S mutation was

constructed to test the effect of preserving a hydrophilic but

uncharged character in the loop region, and the S283C

mutant was a replacement of the serine hydroxyl group with the more nucleophilic thiol group (also potentially capable of subsequent chemical modification, if required)

Effects of mutations on catalytic activity The catalytic activity of E1 mutants was investigated using three different assays: the DCPIP assay, which measures E1 activity by monitoring the reduction of DCPIP as an artificial electron acceptor instead of lipoamide; the reductive acety-lation assay, which measures the rate of reductive acetyacety-lation

of the lipoyl group on a free lipoyl domain; and the PDH assay, which measures the overall activity of a PDH complex reconstituted from E2, E3 and the relevant mutant E1

In all assays, the E1aS283C and E1aF266A mutants behaved essentially the same as wild-type E1 In the DCPIP assay, the E1aY281A and E1aR282A mutants displayed a catalytic activity about twice that of wild-type E1 The single mutants E1aR267A and E1aD276A and the multiple mutants E1aY281A/R282A/S283A and E1aY281S/R282S displayed catalytic activities 2.5 and 3.5 times, respectively, that of wild-type E1 In contrast, the reductive acetylation assay showed no significant changes for the mutants compared with wild-type E1 However, in the PDH assay, the catalytic activity fell to about 50% of the wild-type value for the E1aD276A and E1aR282A mutants and to about 25% for the E1aY281A mutant The double mutants showed even lower (< 20%) activity, but the most

Fig 2 Active site sequence alignment of the E1a chains of 2-OADH complexes The numbering refers to the sequence of the E1a chain from B stearothermophilus (ODPA_BACST) The sequences were taken from the SwissProt and NCBI databases and aligned using

CLUSTALW 1.8 (*), identical residues in sequences in the alignment by

CLUSTALW ; (:), conserved substitutions; (.), semiconserved substitu-tions Residues conserved in all aligned sequences are printed in bold, the conserved sequence motif at the active site is underlined P1 and P2 mark phosphorylation sites 1 and 2 (serine residues) in the E1a chains

of eukaryotic PDH and BCDH complexes ODPA and ODPT, E1a chain of PDH complexes with heterotetrameric E1(a 2 b 2 ); ODBA, E1a chain of heterotetrameric E1(a 2 b 2 ) of BCDH complexes.

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significant drop in PDH complex activity (to < 10%) was

experienced by the E1aR267A mutant (Table 1)

Determination of kinetic parameters

Kinetic parameters for the mutant E1s were determined for

the substrate pyruvate and the cofactor ThDP using the

DCPIP assay (Table 2) All activities recorded were good

fits to Michaelis–Menten kinetics A big increase in the Km

for pyruvate was observed for the E1aR267A mutant, to

nearly 300 lMcompared with about 1 lMfor wild-type E1

The Km for pyruvate was also found to be substantially

increased to more than 50 lMfor E1s carrying mutations at

E1aY281 This was reflected in a major drop in the value of

kcat/Km, in spite of the kcatbeing more than twice that for

wild-type E1 In contrast, the E1aD276A and E1aR282A

mutants more closely resembled wild-type E1, with a Kmfor

pyruvate only about 10 times higher The E1aF266A and

E1aS283C mutants were almost identical to wild-type E1

(Kmfor pyruvate 1.2 lMand 2.1 lM, respectively)

Wild-type E1 was found to have an apparent Kmfor ThDP

of 23 lM Interestingly, the mutants with a significantly

increased Kmfor pyruvate displayed markedly (5-to 20-fold)

lower apparent Kms for ThDP than did wild-type E1 The

E1aR267A mutant was found to have the lowest apparent

Kmfor ThDP (< 1 lM), followed by the E1aY281S/R282S,

E1aY281A/R282A/S283A and E1aY281A/R282A mutants

( 3.0 lM) The single mutations E1aR282A (Km¼

5.2 lM), E1aY281A (Km¼ 5.5 lM) and E1aS283C (Km¼ 13 lM) were less severe in their effects Replacement

of E1aF266 and E1aD276 with alanine had no significant influence on the Kmfor ThDP (Km¼ 21 lM)

Dependence of E1 activity on temperature The temperature-dependence of the catalytic activity of E1 was measured using the DCPIP assay over a range of 25 to

85C (Fig 3) The reaction mixture with E1 was incubated for exactly 10 min at the relevant temperature and the catalytic activity at that temperature was then determined The inactivation temperatures for all the E1 mutants were found to be 5–10C below that of wild-type E1; mutants with a replacement of E1aY281 tended to lose catalytic activity at a slightly lower temperature than mutants retaining that residue The E1aR267A mutant displayed a distinctly lower specific activity at higher temperatures than the other E1a mutants or than wild-type E1 The specific activity of the E1aD276A mutant increased steadily to reach twice that of wild-type E1 at 65C, after which the enzyme was inactivated

Binding of E1 to the peripheral subunit-binding domain of E2

The E1 component of the PDH complex of B stearother-mophilusis bound to the E2 core mainly by interaction of its

Table 2 Kinetic parameters of wild-type and mutant E1s determined by the DCPIP assay.

k cat (s)1) K m (l M )

k cat /K m ( M )1 Æs)1· 10 3 ) k cat (s)1) K m (l M )

k cat /K m ( M )1 Æs)1· 10 3 ) Wild-type 0.48 ± 0.01 1.1 ± 0.1 427 0.46 ± 0.01 23 ± 2 20

E1aF266A 0.30 ± 0.01 1.2 ± 0.1 246 0.35 ± 0.01 21 ± 2 17

E1aR267A 1.62 ± 0.02 291 ± 14 6 1.25 ± 0.01 0.56 ± 0.04 2232

E1aD276A 0.71 ± 0.01 11.1 ± 0.5 64 0.91 ± 0.02 21 ± 2 42

E1aY281A 0.82 ± 0.01 63 ± 3 13 0.97 ± 0.01 5.5 ± 0.3 177

E1aR282A 0.55 ± 0.01 8.8 ± 0.6 63 0.62 ± 0.01 5.2 ± 0.4 121

E1aS283C 0.38 ± 0.01 2.1 ± 0.2 186 0.45 ± 0.01 13 ± 1 33

E1aY281A/R282A/S283A 1.23 ± 0.02 49 ± 4 25 1.39 ± 0.02 2.9 ± 0.2 481

E1aY281A/R282A 0.96 ± 0.01 56 ± 3 17 0.98 ± 0.01 3.0 ± 0.1 326

E1aY281S/R282S 1.21 ± 0.02 66 ± 4 19 1.30 ± 0.01 2.7 ± 0.1 475

Table 1 Specific activities of wild-type and mutant E1s in the various assays.

DCPIP assay Reductive acetylation assay PDH assay

Wild-type 0.128 ± 0.004 100 2.6 ± 0.3 100 12.4 ± 0.3 100

E1aY281A/R282A/S283A 0.452 ± 0.003 354 3.0 ± 0.1 115 1.75 ± 0.06 14 E1aY281A/R282A 0.279 ± 0.004 218 2.2 ± 0.1 85 1.74 ± 0.04 14 E1aY281S/R282S 0.41 ± 0.01 326 2.6 ± 0.1 100 1.83 ± 0.05 15

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E1b chains with the PSBD of the E2 chain To investigate

whether this interaction was impaired by the mutations in

the E1a subunit, non-denaturing PAGE and surface

plasmon resonance (SPR) detection were employed

To assess the binding, mutant E1s were mixed with a

molar excess of di-domain (lipoyl domain and PSBD, joined

by the natural linker region) and submitted to

non-dena-turing PAGE Binding of E1 to the di-domain will create an

E1-di-domain band shifted towards a higher molecular

mass than that of free E1 No difference in the ability of the

mutant E1s to bind to the PSBD was observed (Fig 4A),

though it should be noted that small differences in affinity

would not be detected by this band-shift assay SPR

detection was used to determine the kinetic parameters of

the interaction of the mutant E1s with the PSBD (Fig 4B)

For this purpose, lipoylated di-domain was immobilized on

a BIAcore sensor chip by means of the lipoyl group on the

lipoyl domain (see Materials and methods) E1 was then

injected and allowed to interact with the exposed PSBD of

the di-domain retained on the sensor surface The kon

(association rate constant), k (dissociation rate constant)

and Kd(dissociation constant) of the E1–PSBD interaction were determined The kinetic parameters for the interaction

of wild-type E1 with PSBD reported earlier [35] are: kon, 3.27· 10)6M )1Æs)1; koff, 1.06· 10)3s)1; Kd, 3.24·

10)10M )1 The kinetic constants and Kdvalues for mutant and wild-type E1s were found to be essentially identical (normally within ± 10%)

Discussion Function of the mutated amino acids The E1aY281 and E1aR282 mutants of B stearothermo-philusE1 show a higher Vmaxin the DCPIP assay, no change

in activity in the reductive acetylation assay and a significant drop in overall activity in the PDH assay when compared with wild-type E1 This is similar to the effects on E1 catalysis

of limited proteolysis at Tyr281 and Arg282 [20] Therefore,

Fig 4 Gels and SPR (A) Non-denaturing polyacrylamide gel elec-trophoresis of wild-type and mutant E1s with di-domain at a 16-fold molar excess of di- domain over E1 Lane 1: E1 wild- type; lane 2: E1 wild-type + di-domain; lane 3: E1aF266A mutant; lane 4: E1aF266A mutant + di-domain; lane 5: E1aR267A mutant; lane 6: E1aR267A mutant + di-domain; lane 7: E1aD276A mutant; lane 8: E1aD276A mutant + di-domain The gels for the other mutants were virtually identical (data not shown) (B) SPR sensorgrams for the interaction of wild-type and mutant E1s with the PSBD Shown are the sensorgrams for wild-type E1, the E1aF266A, E1aR267A and E1aD276A mutants The sensorgrams of the other mutants were essentially identical (data not shown).

Fig 3 Temperature-dependence of the activity in the DCPIP assay of

wild-type and mutant E1s.

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it is clear that those effects were not due simply to cleavage of

the Ela chain backbone at these positions

In comparing the results from the different assays, one

should be aware of the differences in reaction conditions

Thus, the reductive acetylation assay is performed with the

lipoyl domain at a concentration of 8.5 lM and E1 at

95 nM, whereas in the PDH assay the local concentrations

of E1 and lipoyl domains in the assembled complex are in

the millimolar range and closer to equimolar This may be

of particular importance as the interaction between E1 and

the lipoyl domain for the E coli PDH complex is estimated

to have a dissociation constant in the millimolar range or

higher despite the fact that the Km is c 20 lM [37,38]

Another difference in the assays is the concentration of the

substrate, pyruvate The different types of assay are reflected

in the different rates; the PDH assay has a turnover number

100-fold higher than the DCPIP assay and 4800-fold higher

than the reductive acetylation assay (referring to wild-type

E1, Table 1) Thus, subtle changes in the kinetics of the E1

reaction may not be detected by the reductive acetylation

assay, but will be detected by the PDH assay

The interaction of E1 (a2b2) with the PSBD was

unaffected by any of the mutations in E1a (Fig 4)

Therefore, any effects of the mutations on the catalytic

activities of the PDH complex must be due to direct effects

on the reactions catalysed by E1 The falls in PDH complex

activity were most marked for the R267A mutant and all the

Y281A mutants (Table 1) Given that there were no

detectable effects on the catalytic activities in the reductive

acetylation assay measured with the free lipoyl domain as

substrate (Table 1) and that the PDH complex activity was

measured with pyruvate at a concentration of 2 mM, well

above the highest Km(300 lM) recorded for any mutant E1

(R267A), the mutations appear to be affecting the ability of

E1 to catalyse the reductive acetylation of the tethered lipoyl

domain in the assembled PDH complex It should be noted

that Tyr281 and Arg282 are located at the mouth of the

funnel-shaped active site of E1, at the bottom of which lies

the ThDP cofactor some 20–25 A˚ from the protein surface

[14] Thus it may be that these mutations are affecting the

efficacy of the recognition of the tethered lipoyl domain by

E1, an essential prelude to reductive acetylation of the

pendant lipoyl group [5,9,39] The damaging effects of

mutations in the b-turn region of the B stearothermophilus

lipoyl domain on its reductive acetylation by native E1 have

been reported earlier [23,40]

The E1aR267A mutant of E1 exhibited a vastly

increased Kmfor pyruvate (300 lM) compared with

wild-type E1 (1 lM) Interaction of the positively charged side

chain of Arg267 with the negatively charged carboxyl

group of the 2-oxo acid substrate thus appears to be very

likely The crystal structures of E1 show the corresponding

arginine residues in P putida [14] and human [15] E1s to be

in a suitable position to participate in such an electrostatic

interaction (Fig 1) In contrast, given the Km(9 lM) for

pyruvate shown by the E1aR282A mutant and its other

modest differences from wild-type E1 (Tables 1 and 2), a

direct role for Arg282 in the E1 reaction appears

improb-able This too is consistent with the E1 crystal structures in

that the arginine residues corresponding to E1aR282 of the

B stearothermophilus E1 are not pointing towards the

active site, identified by the C2-carbon of the cofactor ThDP

Although Arg282 is even more strictly conserved in sequence alignments than Tyr281, the latter is more important for the reaction catalysed by B stearothermophi-lusE1p Apart from the Kmfor pyruvate being markedly increased for all the Tyr281 mutants (Table 2), these enzymes started to lose catalytic activity at lower temper-atures than the other mutants or wild-type E1 (Fig 3) This suggests that Tyr281 has some part to play in conferring thermal stability In the crystal structure of pyruvate decarboxylase (EC 4.1.1.1) from Zymomonas mobilis, a tyrosine sidechain is found in the active site and is suitably oriented to take part in catalysis, as model building with reaction intermediates suggests [41] It was speculated that the tyrosine might form a hydrogen bond with the carboxy group of 2-(2-hydroxypropionyl)-ThDP and contribute to the stabilization of its negative charge The conserved Tyr281 in the B stearothermophilus E1a chain may play a similar role in the E1 component of this and other 2-oxo acid dehydrogenase (Fig 1) complexes

The E1aD276A mutant displayed moderate changes in its kinetic properties compared with wild-type E1, some-what similar to those observed for the R282A mutant Adjacent to E1aD276 in the primary structure of the E1a chain from B stearothermophilus E1p is another aspartate residue, E1aD277 This is replaced by proline in many E1a chains (Fig 2), and has not been examined here However, the corresponding residue, E1aD296 of rat E1b, has been reported to be essential [42], so further experiments may be justified The E1aF266A and E1aS283C mutants behaved almost identically to wild-type E1 in all aspects investigated;

a crucial role for Phe266 and Ser283 in the reaction mechanism can thus be excluded

A common sequence motif The high conservation of amino-acid residues in the sequence spanning positions 255–295 discussed above was noted earlier [43], including the phosphorylation sites P1 and P2 (Fig 2), but no particular role was assigned to them The importance of three residues in the E1a component of rat E1b, namely, E1aR288, E1aH292 and E1aD296 of rat E1b, was recognized by alanine mutagenesis [42] These residues were among 10 in the neighbourhood of phos-phorylation site 1 (E1aS293) that were examined, and their replacement with alanine resulted in totally inactive enzymes They correspond to residues Arg267, His271 and Asp276 in the proposed YR–H–D–YR–DE sequence motif (Fig 2) Our results indicate that Arg267 of

B stearothermophilusE1p is involved in binding the 2-oxo acid substrate and that E1aTyr281 and, to a lesser extent, E1aAsp276 and E1aArg282, have some effect on the decarboxylation of pyruvate and the reductive acetylation

of the tethered lipoyl domain in the active PDH complex (although the replacement of none of these residues caused complete inhibition) Other experiments (M Fries & R N Perham, unpublished work) indicate that E1aHis271 has a crucial part to play in the decarboxylation of pyruvate, probably by serving to stabilize the energetically unfavou-rable dianion formed after nucleophilic attack of ThDP at

Trang 8

the 2-oxo group of the substrate [for a detailed formulation

of the mechanism to date, see [44])

The importance of this region is emphasized by the

reports of clinically deleterious mutations in the human E1a

chain; for example, the replacement of His263 (equivalent to

His271 in B stearothermophilus E1a, Fig 2) with leucine is

associated with a very low PDH complex activity, as is the

replacement of Arg273 (equivalent to Arg282) in B

stearo-thermophilusE1a with cysteine [45,46] It is interesting to

note that the R282A mutation in B stearothermophilus E1a

had only a modest effect on the E1 catalytic activity in vitro

(Tables 1 and 2), perhaps because the amino-acid replacing

the arginine is different

Two phosphorylation sites in eukaryotic E1a chains are

located within the region of the sequence motif outlined in

Fig 2 In addition to the E1aR288A mutant of rat E1b

being inactive, it was incapable of becoming

phosphory-lated, suggesting that this residue may be involved in the

interaction with the E1 kinase [42] Studies on synthetic

peptides as substrates for mammalian pyruvate

dehydro-genase kinase have indicated that an acidic residue to the

C-terminal side of phosphorylation site 1 (Ser293) is also an

important specificity determinant for the kinase [47] This

might be true similarly for phosphorylation site 2 (Ser300)

There are conserved acidic residues to the C-terminal side of

both phosphorylation sites 1 and 2 in the Ela sequence

alignment (Fig 2)

Non-equivalence of active sites has been observed in the

crystal structure of the thiamin-dependent yeast pyruvate

decarboxylase; one active site was found to be in an open

conformation, with two loop regions disordered, whereas in

the other these loop regions were well-ordered and shielded

the active site from the bulk solution [48] The loop housing

the conserved sequence motif in B stearothermophilus E1p

is exposed and flexible, as indicated by limited proteolysis;

moreover, it appears to adopt two different conformations,

one susceptible to proteolysis and the other not [20] Thus,

this loop in the B stearothermophilus E1p might take part in

a similar open-close mechanism If in the mutant E1s the

loop region became more disordered, it might not function

properly as a lid for the active site, thereby granting easier

access for the cofactor ThDP to its binding site The lower

thermal stability of the mutant E1s and the increased Vmax

in the DCPIP assay, which could be due to easier access of

DCPIP to the active site or to a more ready release of

product, would be consistent with this idea

We have looked at the recently determined crystal

structure of the dimeric E1p from the E coli PDH complex

[49] but have been unable to locate a similar sequence of

amino acids in a comparable position with respect to the

active site Some conclusions about the importance of

individual residues in the E coli E1p have been drawn [49],

but a detailed comparison of the two types of E1 (dimeric

and heterotetrametric) will have to be the subject of further

investigation

Acknowledgements

This work was supported by a research grant from the Biotechnology

and Biological Sciences Research Council (to R N P.) M F is

grateful to the Verband der Chemischen Industrie and the

Cusanu-swerk for a studentship and Clare Hall for additional support, H J C.

thanks the BBSRC for the award of an earmarked Research Studentship, and H.-I J is grateful to the Cambridge Overseas Trust,

St John’s College, Cambridge and the Department of Biochemistry, University of Cambridge for financial support.

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