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Unlike prokaryotic ABC proteins, NBD1 and NBD2 of SUR1 show significant sequence differences: thus, the ATPase activity of the isolated recombinant NBD homodimers will not necessarily refl

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Heidi de Wet1, Michael V Mikhailov1, Constantina Fotinou1, Mathias Dreger1, Tim J Craig1,

Catherine Ve´nien-Bryan2and Frances M Ashcroft1

1 Henry Wellcome Centre for Gene Function, Department of Physiology, Anatomy and Genetics, University of Oxford, UK

2 Laboratory of Molecular Biophysics, Department of Biochemistry, University of Oxford, UK

ATP-sensitive potassium (KATP) channels couple cell

metabolism to membrane excitability and

transmem-brane ion fluxes In pancreatic b-cells, they are of

crucial importance for regulating insulin secretion [1]

At substimulatory glucose concentrations, KATP chan-nels are open and generate a negative potential that

Keywords

ABCC8; ATPase activity; K ATP channel;

nucleotide-binding domain; sulfonylurea

receptor

Correspondence

F M Ashcroft, University Laboratory of

Physiology, Parks Road, Oxford OX1 3PT,

UK

Fax: +44 1865 285812

Tel: +44 1865 285810

E-mail: frances.ashcroft@physiol.ox.ac.uk

(Received 4 April 2007, revised 8 May 2007,

accepted 14 May 2007)

doi:10.1111/j.1742-4658.2007.05879.x

The ATP-sensitive potassium (KATP) channel couples glucose metabolism

to insulin secretion in pancreatic b-cells It comprises regulatory sulfonyl-urea receptor 1 and pore-forming Kir6.2 subunits Binding and⁄ or hydro-lysis of Mg-nucleotides at the nucleotide-binding domains of sulfonylurea receptor 1 stimulates channel opening and leads to membrane hyperpolari-zation and inhibition of insulin secretion We report here the first purifica-tion and funcpurifica-tional characterizapurifica-tion of sulfonylurea receptor 1 We also compared the ATPase activity of sulfonylurea receptor 1 with that of the isolated nucleotide-binding domains (fused to maltose-binding protein to improve solubility) Electron microscopy showed that nucleotide-binding domains purified as ring-like complexes corresponding to  8 momomers The ATPase activities expressed as maximal turnover rate [in nmol

PiÆs)1Æ(nmol protein))1] were 0.03, 0.03, 0.13 and 0.08 for sulfonylurea receptor 1, nucleotide-binding domain 1, nucleotide-binding domain 2 and

a mixture of nucleotide-binding domain 1 and nucleotide-binding domain 2, respectively Corresponding Km values (in mm) were 0.1, 0.6, 0.65 and 0.56, respectively Thus sulfonylurea receptor 1 has a lower Km than either of the isolated nucleotide-binding domains, and a lower max-imal turnover rate than nucleotide-binding domain 2 Similar results were found with GTP, but the Kmvalues were lower Mutation of the Walker A lysine in nucleotide-binding domain 1 (K719A) or nucleotide-binding domain 2 (K1385M) inhibited the ATPase activity of sulfonylurea recep-tor 1 by 60% and 80%, respectively Beryllium fluoride (Ki16 lm), but not MgADP, inhibited the ATPase activity of sulfonylurea receptor 1 In con-trast, both MgADP and beryllium fluoride inhibited the ATPase activity of the nucleotide-binding domains These data demonstrate that the ATPase activity of sulfonylurea receptor 1 differs from that of the isolated nucleo-tide-binding domains, suggesting that the transmembrane domains may influence the activity of the protein

Abbreviations

ABC, ATP-binding cassette; BeF, beryllium fluoride; CFTR, cystic fibrosis transmembrane conductance regulator; DDM, dodecylmaltoside; DMPC, 1,2-dimyristoyl-sn-glycero-phosphocholine; EM, electron microscopy; KATP, ATP-sensitive potassium; MBP, maltose-binding protein; MRP1, multidrug resistance protein 1; NBD, nucleotide-binding domain; SUR, sulfonylurea receptor; SUR1F, full-length flagged-tagged SUR1;

WA, Walker A; WB, Walker B.

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keeps voltage-gated Ca2+ channels closed and

abol-ishes Ca2+ influx Because a rise in intracellular Ca2+

is needed to stimulate insulin granule release, this

pre-vents insulin secretion When plasma glucose levels

increase, glucose uptake and metabolism lead to

chan-ges in the intracellular concentrations of adenine

nucleotides that close KATP channels, triggering Ca2+

channel opening, Ca2+influx, elevation of intracellular

Ca2+and insulin release

The b-cell KATP channel is a large octameric

com-plex that comprises a central tetrameric Kir6.2 pore

surrounded by four sulfonylurea receptor (SUR) 1

subunits [2] Both Kir6.2 and SUR1 subunits are

involved in the metabolic regulation of channel

activ-ity: ATP binding to Kir6.2 causes channel inhibition

[3], whereas interaction of Mg-nucleotides (MgATP

and MgADP) with SUR1 stimulates channel opening

[4–6] Impairment of nucleotide interactions with either

subunit can lead to neonatal diabetes or its converse,

congenital hyperinsulinism [1]

SUR belongs to the ATP-binding cassette (ABC)

protein superfamily [7] It has 17 transmembrane

heli-ces and two large cytosolic loops, which contain the

nucleotide-binding domains (NBDs) NBD1 and

NBD2 As in all ABC proteins, each NBD contains a

highly conserved Walker A (WA) and Walker B (WB)

motif involved in ATP binding and hydrolysis, an

invariant ‘signature sequence’, and several other

con-served residues Crystallization of a number of

pro-karyotic NBDs and ABC proteins indicates that they

associate in a sandwich dimer conformation [8–11], in

which residues from the WA and WB motifs of one

NBD interact with the signature sequence of the other

NBD to form separate ATP-binding sites, with distinct

properties Each ATP-binding site therefore contains

contributions from both NBD1 and NBD2 Evidence

of physical interaction between the NBDs, and

molecular modeling studies, support the idea that

SUR1 also conforms to the sandwich dimer model

[12,13] Functional studies demonstrate that formation

of such a sandwich dimer is critical for driving gating

of cystic fibrosis transmembrane conductance regulator

(CFTR) channels [14], but this has not yet been

dem-onstrated for KATPchannels

There are two genes that encode SUR, ABCC8

(SUR1) and ABCC9 (SUR2) [15–17] The latter exists

in several splice variants, the most important being

SUR2A and SUR2B Differences in the SUR subunit

contribute to the variable metabolic sensitivities of

KATP channels in different tissues For example, even

when heterologously expressed in the same cell,

recom-binant Kir6.2–SUR2A channels open less readily on

metabolic inhibition than Kir6.2–SUR1 channels [18]

It has been suggested that this may relate to differ-ences in the ATPase activity of SUR1 and SUR2 [19] The ATPase activity of full-length SUR1 has not been measured directly to date However, MgATP hydrolysis has been measured directly for recombinant proteins in which either NBD1 or NBD2 of SUR was fused to the maltose-binding protein [19–21] ATPase activity of native SUR (i.e containing both NBDs and transmembrane domains) has also been inferred by comparing covalent labeling with 8-azido-[32P]ATP[aP] and 8-azido-[32P]ATP[cP] [22] In these studies, how-ever, hydrolysis by NBD2, but not NBD1, was detec-ted Unlike prokaryotic ABC proteins, NBD1 and NBD2 of SUR1 show significant sequence differences: thus, the ATPase activity of the isolated recombinant NBD homodimers will not necessarily reflect that of the NBD heterodimer expected for native SUR1 Fur-thermore, the presence of the transmembrane domains

in SUR1 may influence ATPase activity We have therefore purified SUR1 and compared its capacity to hydrolyze ATP and GTP with that of isolated NBD1,

or NBD2, of SUR1 [fused to maltose-binding protein (MBP)] We also measured the ATPase activity of a mixture of NBD1 and NBD2 proteins In addition, the effects of the inhibitors beryllium fluoride (BeF) and MgADP were explored

Results

Purification and characterization of SUR1 MBP–NBDs and SUR1F

SDS⁄ PAGE and Coomassie staining revealed a single major band following purification of full-length flagged-tagged SUR1 (SUR1F), MBP–NBD1 and MBP–NBD2 (Fig 1A) For simplicity, we refer to these proteins subsequently as SUR1, NBD1 and NBD2 MALDI-TOF analysis of total purified pro-teins confirmed their identities as well as the absence

of any other contaminating ATPases (data not shown) Additional bands visible on these gels were identified

as degradation products by MALDI-TOF analysis of gel cut-outs

Gel filtration of SUR1 yielded two fractions (Fig 1Ba) The smaller peak corresponds to the molecular mass expected for monomeric SUR1 and the larger peak runs as expected for a mixture of tetra-meric and oligotetra-meric species

Gel filtration revealed that NBD1, NBD2 or a 1 : 1 mixture of NBD1 and NBD2 eluted as a single sharp peak corresponding to a single oligomeric species (Fig 1Bb) Calculated molecular masses gave approxi-mate sizes of 8 monomers for NBD1 and  9

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mono-mers for NBD2 No larger aggregates or other protein

species were detected SDS⁄ PAGE analysis of the

pro-teins in the respective gel filtration fractions confirmed

their identities as MBP–NBD1 and MBP–NBD2

Because gel filtration suggested that NBD1 and NBD2

associate as oligomers, we collected the peak eluates

and examined them by negative stain electron

micros-copy (EM) This revealed that both proteins formed

ring-like oligomers For NBD1, the outer diameter of

the projected structure was between 120 and 140 A˚

and the inner diameter was 40–75 A˚ (Fig 1C) A

sim-ilar structure was observed for NBD2 and for a 1 : 1

mixture of NBD1 and NBD2 Oligomerization was

independent of the presence of MgATP (data not

shown) MBP alone did not form ring-like oligomers

(data not shown), suggesting that the interaction is mediated by the NBD part, rather than the MBP part,

of the MBP–NBD fusion proteins

Nucleotide hydrolysis by SUR1 Recombinant full-length SUR1 hydrolyzed MgATP very slowly, with a maximal turnover rate of 0.03 s)1 (Table 1 and Fig 2A) and a Km of 0.1 mm

No ATPase activity was detected in the absence of

Mg2+ or from protein purified from cells transfected with an SUR1 construct lacking the FLAG tag used for affinity purification (Fig 2A) Because KATP channels are stimulated by GTP, via interaction with the NBDs

of SUR1 [32], we also investigated the ability of SUR1

Octamer Tetramer

Monomer

Oligomer

A

250

150

100

75

50

kDa

1 2

SUR1

188 98

62 49 38 28

kDa

1 2 3

NBD1 NBD2

Fig 1 Purification of SUR1, NBD1 and NBD2 (A) Coomassie-stained denaturing gels of purified SUR1 (left, lane 2), MBP– NBD1 (right, lane 1), and MBP–NBD2 (right, lane 2) Molecular mass markers, lane 1 (left) and lane 3 (right) Samples shown are the purified eluates from affinity resins, and and were not subjected to further purifica-tion by gel filtrapurifica-tion (Ba) Gel filtrapurifica-tion analy-sis of purified SUR1 (Bb) Gel filtration analysis of purified MBP–NBD1 (Ca) Negat-ive stain electron micrograph of MBP– NBD1 The scale bar is 100 nm Black arrows indicate ring-like structures The white arrow points to a stack of rings (Cb) Ten different classes of particle The size of the boxes is 280 A ˚

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to hydrolyze GTP Figure 2B shows that GTP was also

hydrolyzed, but with a much higher Km (> 1 mm),

which suggests that GTP binds to SUR1 with a lower

affinity than ATP The turnover rate, estimated by

fit-ting the data to the Michaelis–Menten equation, was similar to that of ATP

Mutation of residues in the Walker motifs of SUR impairs channel activation by MgATP and MgADP [5] Mutating the WA lysine in NBD1 of SUR1 to alanine (K719A) reduced ATPase activity by approxi-mately 60%, whereas mutating the WA lysine in NBD2 to methionine (K1385M) inhibited ATPase activity by about 80% (Fig 2C) when compared to wild-type controls assayed in parallel (n¼ 2) Neither mutation affected the Km for ATP These data are consistent with the idea that these mutations affect the rate of hydrolysis but do not influence ATP binding

Table 1 ATPase activities and kinetic constants.

Construct

Turnover rate

(nmol P i Æs)1Æ

nmol)1protein)

V max

(nmol P i Æmin)1Æ

mg)1)

K m

MBP–NBD1 0.03 ± 0.003 23.8 ± 2.40 0.6 ± 0.09 14

MBP–NBD2 0.13 ± 0.01 103.81 ± 8.70 0.65 ± 0.13 12

MBP–NBD1 + 2 0.08 ± 0.01 61.22 ± 6.78 0.56 ± 0.11 10

[ATP] (m M )

[ATP] (m M )

Wt of KA KM KA

Wt of KM

[inhibitor] (m M ) 0.001 0.01 0.1 1 0

0.0 0.2 0.4 0.6 0.8 1.0 1.2

0.0

0.2

0.4

0.6

0.8

1.0

A

0

2

4

6

8

10

12

MgATP

No FLAG tag

Mg free

C

D

[GTP] (m M )

0 2 4 6 8 10

12

B

Fig 2 ATPase activity of SUR1 (A) ATPase activity in the presence (filled circles, n ¼ 6) or absence of Mg2+(crosses, n ¼ 2) of purified SUR1, at 37 C Membranes from cells expressing SUR1 lacking a FLAG tag and purified as usual show no ATPase activity (open circles,

n ¼ 1) (B) GTPase activity of SUR1 (n ¼ 4) The line is fitted to the Michaelis–Menten equation, with an estimated V max and Kmof 10 nmol

P i Æmin)1Æmg)1and 0.86 m M , respectively (C) ATPase activity of SUR1 containing the mutations K719A (triangles, n ¼ 2) or K1385M (dia-monds, n ¼ 2) Data are expressed as a fraction of the turnover rate for wild-type SUR1 assayed in parallel (D) Inhibition of ATPase activity

at 1 m M MgATP by ADP (open circles, n ¼ 4) and by BeF (filled circles, n ¼ 3) Data are expressed as a fraction of the turnover rate in the absence of inhibitor.

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BeF (BeF3 and BeF42–) is a potent inhibitor of

ATP hydrolysis by many ABC proteins, including

P-glycoprotein [33] and the isolated NBD2 of SUR2A

[20] It acts by arresting the ATPase cycle in the

pre-hydrolytic conformation [34] The ATPase activity of

SUR1 was potently and completely inhibited by BeF

(Ki16 lm; Table 2; Fig 2D) Previous studies have

shown that MgADP blocks the ATPase activity of

NBD2 of SUR2A [20] by trapping the ATPase cycle in

the posthydrolytic conformation Unexpectedly,

how-ever, no effect of ADP on the ATPase activity of

SUR1 was observed (Fig 2D)

The ATPase activity of SUR1 is about 10-fold less

than what we previously reported for the complete

octameric KATP channel complex, SUR1–Kir6.2

(turn-over rate 0.4 ± 0.03 s)1 calculated from Mikhailov

et al [2])

Nucleotide hydrolysis activities of NBD1 NBD1 displayed low maximal ATPase activity, similar

to that of SUR1, but the Kmwas about six-fold larger (0.6 mm, P < 0.005) (Table 1 and Fig 3A) This sug-gests that ATP binds more tightly to full-length SUR1 than to the isolated NBD1 GTP was hydrolyzed with

a Kmhigher than that for ATP (Fig 3B) There was a very small, but significant, apparent hydrolysis of ADP (Table 3, Fig 3B) Negligible ATP hydrolysis was observed in the absence of Mg2+ or in protein prepa-rations from cells expressing MBP alone (Fig 3A) BeF blocked ATP hydrolysis at NBD1 with a

Ki of 33 lm (Table 2 and Fig 3C) Unlike with SUR1, however, inhibition appeared to be incom-plete, and the maximal block was 76% In marked contrast to SUR1, MgADP inhibited ATP hydrolysis

[Nucleotide] (m M )

0 5 10 15 20 25 30

ADP GTP

[ATP] (m M )

0

5

10

15

20

25

A

C

B

[Inhibitor] (m M )

0.0

0.2

0.4

0.6

0.8

1.0

1.2

BeF

ADP

MBP

–Mg2+

+Mg2+

Fig 3 ATPase activity of NBD1 (A) ATPase activity of NBD1 in the presence (filled circles, n ¼ 14) or absence (open circles,

n ¼ 2) of Mg 2+ , at 37 C MBP control (open triangles, n ¼ 2) (B) GTP (filled circles, n ¼ 3) and ADP (filled triangles, n ¼ 3) hydrolytic activity The line is fitted to the Michaelis–Menten equation, through the GTP data points with an estimated Vmaxand

Kmof 39 nmol PiÆmin)1Æmg)1and 2.6 m M , respectively (C) Inhibition of ATPase activity

at 1 m M MgATP by ADP (open circles, n ¼ 3) or BeF (filled triangles, n ¼ 3) Data are expressed as a fraction of the turnover rate

in the absence of inhibitor.

Table 2 Inhibition constants for ADP and BeF ND, not detected n indicates the number of different protein preparations.

Construct

IC 50 (ADP) (m M )

K i (ADP)

IC 50 (ADP) (m M )

K i (BeF)

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at NBD1, with a Ki of 2.1 mm (Table 2 and

Fig 2C)

Nucleotide hydrolysis activities of NBD2

The maximal ATPase activity of NBD2 was about

four-fold greater than that of either SUR1

(P < 0.001) or NBD1 (P < 0.001) The Km was

sim-ilar to that of NBD1 and about six-fold larger than

that of SUR1 (Table 1, Fig 4A (P < 0.005) These

results suggest that the ATP-binding affinities of

NBD1 and NBD2 are similar, but that the hydrolytic

step occurs more rapidly in NBD2, and that,

com-pared with SUR1, the ATP-binding affinity of NBD2

is less but hydrolysis is faster GTP was also

hydro-lyzed, with a Km higher than that for ATP (Fig 4B)

MgADP was hydrolyzed at very low rate, but this was

about three-fold greater than that of NBD1 (Table 3, Fig 4B) Negligible ATP hydrolysis was observed in the absence of Mg2+

As found for NBD1, MgATP hydrolysis (1 mm) was inhibited by both BeF and MgADP (Table 2 and Fig 4C) The Kifor BeF inhibition (19 lm) was lower than that for NBD1, and maximal inhibition was 86% ATP hydrolysis was also inhibited by MgADP, with a Kiof 0.84 mm (Table 2)

Nucleotide hydrolysis activities of NBD1 + NBD2 Interactions of the isolated MBP–NBDs of the CFTR, or the multidrug resistance protein MRP1, have been demonstrated previously [35,36] We there-fore examined ATP hydrolysis in a 1 : 1 mixture of

NBD1 + NBD2 mixture had a maximal turnover rate intermediate between that of NBD1 and NBD2 alone, and the Km(0.56 mm) was not significantly dif-ferent from that of either NBD1 or NBD2 (Table 1, Fig 5A) Thus, as found for MRP1 [36], mixing the two NBDs did not have a major impact on the cata-lytic activity of either NBD

GTP was hydrolyzed by the NBD1 + NBD2 mix-ture with Km> 1 mm (Fig 5B) As observed for the

[ATP] (m M )

0 20 40 60 80 100 120 140

A

C

B

[Nucleotide] (m M )

0 20 40 60 80 100

120 GTP ADP

[Inhibitor] (m M )

0.0 0.2 0.4 0.6 0.8 1.0 1.2

ADP BeF

-Mg2+

+Mg2+

Fig 4 ATPase activity of NBD2 (A) ATPase

activity of NBD2 in the presence (filled

cir-cles, n ¼ 12) or absence (open circles, n ¼

2) of Mg 2+ , at 37 C (B) GTP (filled circles,

n ¼ 3) and ADP (filled triangles, n ¼ 3)

hydrolytic activity The line is fitted to the

Michaelis–Menten equation, with an

estima-ted Vmaxand Kmof 153 nmol PiÆmin)1Æmg)1

and 2.2 m M , respectively (C) Inhibition of

ATPase activity at 1 m M MgATP by ADP

(open circles, n ¼ 3) or BeF (filled triangles,

n ¼ 4) Data are expressed as a fraction of

the turnover rate in the absence of inhibitor.

Table 3 ADPase activities and kinetic constants.

Construct

Turnover rate

(nmol P i Æs)1Æ

nmol)1protein)

V max

(nmol P i Æ min)1Æmg)1)

K m

MBP–NBD1 0.005 ± 0.001 3.83 ± 1.26 0.17 ± 0.08 3

MBP–NBD2 0.014 ± 0.003 11.26 ± 2.40 0.07 ± 0.02 3

MBP–NBD1 + 2 0.012 ± 0.002 9.80 ± 2.69 0.06 ± 0.01 3

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individual domains, the NBD mixture apparently

hydrolyzed MgADP but with a very low turnover rate

(Table 3, Fig 5B), and no hydrolysis was observed in

the absence of Mg2+

MgATP hydrolysis was inhibited by BeF with a Ki

of 20 lm (Table 2, Fig 5C), which is not significantly

different from that of either NBD1 (30 lm) or

NBD2 (19 lm) alone However, the Ki for MgADP

inhibition of ATP hydrolysis was significantly less

than that of NBD1 (P < 0.05): it was also lower than

that of NBD2, although this difference was not

significant (Table 2) A value intermediate between

those of NBD1 and NBD2 would be expected if

the NBDs were functionally independent: thus, these

data suggest that the NBDs may functionally interact

when mixed This finding is consistent with the

idea that at least some heterodimers of NBD1

and NBD2 are present in the NBD1 + NBD2 mixture

Discussion

Nucleotide handling by SUR1

The ATPase activity of SUR1 was 10-fold lower, and

the Km three-fold larger, than that measured for the

purified complete KATP channel (Kir6.2–SUR1)

com-plex [2] This suggests that ATP binds with lower

affinity and the rate of ATP hydrolysis is faster in the KATP channel complex than in SUR1 alone CryoEM analysis revealed that the KATP channel associates as a large octameric complex in which the individual SUR1 subunits are tightly packed around

a central Kir6.2 tetrameric pore [2] The higher ATPase activity of the KATP channel complex might therefore result from interactions between adjacent SUR1 subunits, and⁄ or between SUR1 and Kir6.2, that enhance cooperativity and⁄ or crosstalk between the NBDs

The ATPase activity of purified SUR1 (Vmax,

9 nmol PiÆmin)1Æmg)1) is at the lower end of the range found for MRP1: from 5 to 10 nmol PiÆ min)1Æmg)1 [37] to 470 nmol PiÆmin)1Æmg)1 [38] It is less than that reported for CFTR (60 nmol PiÆ min)1 Æmg)1 protein [39]), or P-glcyoprotein (320–3900 nmol PiÆ min)1Æmg)1 [40,41]), but higher than that found for ABCR (1.3 mol PiÆmin)1Æmg)1 [42]) It is possible that the lower ATPase activity of SUR1 is related to the unique role of this ABC protein as a channel regulator rather than a transporter It is worth noting that the activity of other ABC transporters, including the closely related MRP1 [38], are stimulated by their substrates It is possible that mechanism by which Kir6.2 stimulates the ATPase activity of SUR1 resem-bles this substrate activation

0

20

40

60

80

A

C

B

0 10 20 30 40 50 60

[Inhibitor] (m M )

0.0

0.2

0.4

0.6

0.8

1.0

1.2

ADP

ADP GTP

BeF

Fig 5 ATPase activity of NBD1 + NBD2 (A) ATPase activity of the NBD1 + NBD2 mixture in the presence (filled circles, n ¼ 10) or absence (open circles, n ¼ 2) of

Mg2+, at 37 C (B) GTP (circles, n ¼ 3) and ADP (triangles, n ¼ 3) hydrolytic activity of NBD1 + NBD2 The line through the GTP data points is fitted to the Michaelis– Menten equation, with an estimated Vmax and Kmof 70 nmol PiÆmin)1Æmg)1and 1.7 m M , respectively (C) Inhibition of ATPase activity at 1 m M MgATP by ADP (open circles, n ¼ 3) or BeF (filled triangles,

n ¼ 4) Data are expressed as a fraction of the turnover rate in the absence of inhibitor.

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The Km for ATP hydrolysis by SUR1 (0.1 mm) was

similar to that reported for purified CFTR and MRP1

(0.1–3 mm) [37–39], but significantly lower than we

measured for the isolated NBDs of SUR1 We

specu-late that the presence of the transmembrane domains in

SUR1 induces conformational changes in the NBDs, or

in their association, that influences ATP binding The

Km of the purified KATP channel complex (SUR1F–

Kir6.2) was 0.4 ± 0.2 mm [2], which is not significantly

different from that found for the isolated NBDs, but is

somewhat greater than that of SUR1 Thus, it seems

possible that the presence of Kir6.2 within the KATP

complex may further modify interactions between the

NBDs that occur in SUR1 alone Both the NBDs and

the transmembrane domains of SUR1 are known to

interact with the cytosolic and transmembrane domains

of Kir6.2, respectively [23,25,43]

Mutation of the WA lysines (K719A, K1385M)

reduced ATPase activity by 70–80% Mutation of the

equivalent residues in full-length CFTR [44], or the

isolated NBD2 of SUR2A [45] and NBD1 or NBD2

of CFTR [46, 47], also reduces, but does not fully

abolish, ATPase activity Nevertheless, these mutations

completely ablate the ability of MgADP to stimulate

KATP channel activity [5] Thus, WA mutations in

SUR1 may also influence nucleotide binding [48]

and⁄ or the mechanism by which nucleotide

bind-ing⁄ hydrolysis is coupled to channel activity

ATPase activity of the isolated NBDs

As previously reported, MBP-fusion proteins of

isola-ted NBD1 and NBD2 domains hydrolyzed ATP The

Kmfor ATP hydrolysis ( 600 lm) did not vary

signi-ficantly between the isolated NBDs (NBD1, NBD2 or

the NBD1–NBD2 mixture) Previous studies yielded

somewhat lower values of 290 lm and 350 lm for

NBD1 and NBD2, respectively [19] For comparison,

values for the NBDS of SUR2A were 220 lm for

NBD1 [19] and ranged from 370 lm [19] to 4.4 mm

[45] for NBD2 The rates of ATP hydrolysis that we

observed are about two-fold (NBD1) and up to

five-fold (NBD2) higher than those previously reported for

the isolated NBDs of SUR1 [19] It is possible that

these differences reflect differences in the sequence of

isolated domains used in the different studies Mixing

NBD1 and NBD2 did not alter ATPase activity This

is similar to what is found for MRP1 [36], the ABC

protein most closely related to SUR1, but contrasts

with the NBDs of CFTR, where the activity of NBD1

is enhanced by heterodimerization with NBD2 [35,49]

Like other ABC proteins, including MRP1 [38],

SUR1 and both of its isolated NBDs had a broad

nuc-leotide specificity and hydrolyzed GTP as well as ATP There appeared to be a small amount of hydrolysis of MgADP by both NBD1 and NBD2, which contributed less than 10% of the ATP hydrolysis rate It is possible that SUR1 exhibits adenylate kinase activity, as has been suggested for CFTR [50,51] In this case, hydroly-sis of ATP generated from ADP (by adenylate kinase activity) might account for the increase in free phos-phate that we observed

Inhibition of ATPase activity

A decrease in the ATPase activity of SUR1 was observed when the conserved lysine in the WA motif was mutated either in NBD1 or NBD2 Mutation of the WA motif in NBD1 reduced ATPase activity by about 60% If we assume that the relative extent of ATPase activity at NBD1 and NBD2 remains the same in full-length SUR1 (i.e that of NBD1 is

 20% of that of NBD2), then the marked inhibition

of ATPase activity of SUR1 suggests that the WA mutation in NBD1 also reduced hydrolysis at NBD2 This might indicate possible interactions between the NBDs The fact that the same mutations did not affect MgADP binding to NBD2 [52] suggests that it

is the hydrolytic capacity that is affected Mutation

of the WA lysine at NBD2 blocked ATPase activity

of SUR1 by about 80% Although this would be con-sistent with inhibition of NBD2 alone, it may also reflect a partial decrease in hydrolysis at both NBD1 and NBD2

The ATPase activity of SUR1 was potently inhibited

by BeF, which traps ABC proteins in a prehydrolytic ATP-bound conformation [33,34] Inhibition by BeF (1 mm) has previously only been reported for the iso-lated NBD2 of SUR2A [20,21]

MgADP also inhibited ATP hydrolysis by isolated NBDs, albeit with very low affinity The lowest value

of Ki (0.6 mm) was found for the NBD1 + NBD2 mixture The inability of ADP to block ATP hydroly-sis by SUR1 is surprising: possible explanations for this finding include a lower ADP affinity for SUR1 or

a higher adenylate kinase activity We presume that this effect is ameliorated in the KATPchannel complex,

as MgADP stimulates channel activity, and reverses channel inhibition by ATP, via interaction with the NBDs of SUR1 [4,5]

Oligomerization of the NBDs Gel filtration indicated that MBP–NBD1, MBP–NBD2 and a 1 : 1 mixture of the two purified as a multimer

of around eight or nine monomers When viewed by

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EM, the proteins formed ring-like structures with an

outer diameter of  120–140 A˚ This is similar to the

outer diameter of the purified octameric KATPcomplex

(180 A˚) [2], and is consistent with the idea that the

ring-like structures represent eight MBP–NBDs that

coassemble as a tetramer of dimers The inner diameter

of the NBD ring was 40–75 A˚ This space is expected

to be occupied by Kir6.2 in the native KATP channel

complex The widest diameter of cytoplasmic domain

of the related Kir channels Kir3.1 and Kir3.2 was

 80 A˚ in the crystal structure [53,54] Thus, the NBDs

are likely to pack somewhat less tightly in the KATP

complex than in the ring-like structures that we

observed for the isolated NBDs

These results suggest that the NBDs may be involved

in physical subunit–subunit associations within the

KATP channel complex, and raise the possibility that

they may also be involved in functional interactions

between subunits Previous studies have also suggested

that NBD1 and NBD2 can physically interact [23,24]

and that purified NBD1 of SUR1 can exist as a tetramer

[26] Interaction of isolated recombinant NBDs to form

functional heterodimers has also been reported for

several other ABC proteins [35,36,55] Such

hetero-dimerization enhanced the ATPase activity of some

ABCC proteins (e.g CFTR) [35,48], attenuated ATPase

activity in others (e.g ABCR [55]), or was without effect

(e.g MRP1 [36]), as we found for SUR1

Implications for channel gating

Unlike those of other ABC proteins, the functional

role of SUR1 is that of a channel regulator, and ATP

hydrolysis by SUR1 plays an important role in the

metabolic regulation of the KATP channel [23] In

electrophysiologic studies, both MgATP and MgADP

stimulate KATP channel activity [3–6] However,

cur-rent evidence suggests that it is the presence of

MgADP at NBD2 that results in KATPchannel

open-ing, and that MgATP must be hydrolyzed to MgADP

in order for channel activation to occur [20]

It is difficult to measure the EC50 for MgATP

acti-vation of wild-type KATP currents in

electrophysiolog-ical studies, due to simultaneous inhibition via the

ATP-binding site of Kir6.2 Coexpression of SUR1

with Kir6.2 carrying mutations in the ATP-binding

site, however, suggests that half-maximal channel

acti-vation is produced by MgATP concentrations of

around 0.1 mm or greater [6] This is in agreement

with the results we report here for SUR1 and those

found previously for the KATPcomplex [2]

Mutation of the WA lysines markedly decreased but

did not completely abolish ATP hydrolysis by SUR1,

in agreement with the electrophysiological data The same mutations shifted the IC50for ATP block of the

KATP channel to a value (13–16 lm) [6]) intermediate between that seen for wild-type channels in the pres-ence ( 30 lm) [6] and abspres-ence (6 lm) [6] of Mg2+ One might expect that a mutation which abolished MgATP binding⁄ hydrolysis would have an IC50similar

to that found in Mg-free solution for wild-type chan-nels The fact that this is not the case suggests that binding⁄ hydrolysis of MgATP is not entirely abolished

by WA mutations Interestingly, the same mutations completely abolished the ability of MgADP to stimu-late KATPchannel currents [5]

Conclusion

SUR1 is unique among ABC proteins in that it serves

as a channel regulator, forming a tightly associated octameric KATPchannel complex in which four Kir6.2 subunits form a central pore surrounded by four SUR1 subunits [2] The fact that the isolated NBDs of SUR1 associate in tetrameric ring-like structures even when Kir6.2 is not present suggests that these domains possess some intrinsic capacity for stable association and that this may contribute to formation of the octa-meric KATP channel complex Here we show that the ATPase activity of SUR1 alone differs from those of both the isolated NBDs and of the octameric KATP channel complex This suggests that the ATPase activ-ity of the NBDs is influenced both by the presence of the transmembrane domains of SUR1 and by the tetrameric Kir6.2 pore Thus, just as SUR1 influences the channel activity of Kir6.2, so Kir6.2 appears to modulate the ATPase activity of SUR This may be considered analogous to the way in which substrates stimulate the activity of other ABC proteins

Experimental procedures

Protein expression and purification

A FLAG-tag was inserted into the extracellular loop between transmembrane helices 11 and 12 of rat SUR1 (GenBank L40624), as previously reported [2] This full-length construct of SUR1 (SUR1F) was expressed in insect cells (Sf9) using a baculovirus expression system (Invitrogen, Paisley, UK), and expression was verified and quantified by [3H]glibenclamide binding [23] Cells were grown and harvested as previously described [2], dis-rupted using a Stansted TC5W homogenizer (Stansted Fluid Power Ltd, Stansted, UK) at a pressure of

10 000 lbÆin)2, and centrifuged at 200 g for 10 min using

a Beckman Allegra 6KR centrifuge with S/N02E3297

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rotor The supernatant was loaded on a step sucrose

gra-dient (10%⁄ 46%) and centrifuged at 100 000 g for 1 h

using a Beckman L7 centrifuge with SW28 rotor The

intermediate phase was collected and diluted four times

with 50 mm Tris⁄ HCl (pH 8.8) and 200 mm NaCl

Do-decylmaltoside (DDM) (0.5% w⁄ v) was then added, and

membranes were solubilized for 20 min at room

tempera-ture, and then centrifuged at 48 400 g for 20 min using a

Beckman Avanti J-20XP centrifuge with JA-25.50 rotor

Anti-FLAG M2 affinity gel (Sigma, Poole, UK) was

added to the supernatant and incubated for 2 h The

suspension was washed with 20 volumes of 50 mm

Tris⁄ HCl (pH 8.8), 150 mm NaCl and 0.1% DDM

Pro-tein was then eluted with 100 lm 3-FLAG peptide

(Sigma), 50 mm Tris⁄ HCl (pH 8.8), 150 mm NaCl, 0.2%

DDM, 0.05% 1,2-dimyristoyl-sn-glycero-phosphocholine

(DMPC) All procedures were carried out at 4C The

purified protein yield ranged between 50 and 100 lgÆL)1

The identity and purity of SUR1F was confirmed by

MALDI-TOF MS All assays were performed on freshly

prepared SUR1F

Rat NBDs were cloned into the pMAL-c2X vector (New

England Biolabs, Hitchin, UK) to yield MBP-fusion

con-structs, in which MBP is attached to the N-terminal end of

the NBD This strategy was employed because the NBDs

alone are known to be poorly soluble [24] The nucleotide

sequence used for NBD1 was Val608 to Leu1004, and that

used for NBD2 was Lys1319 to Lys1581 Plasmids were

transformed into BL21-CodonPlus Escherichia coli cells

(Stratagene, La Jolla, CA, USA) One liter of Terrific Broth

(Sigma) in baffled flasks was inoculated with 50 mL of

transformed BL21-CodonPlus, grown to a D600 of 1 and

induced with 0.4 mm isopropyl thio-b-d-galactoside Cells

were harvested after 4 h, at 200 g for 20 min They were

re-suspended in 30 mL of buffer A (50 mm Tris⁄ HCl, pH 7.5,

150 mm NaCl, 2 mm dithiothreitol and 1% protease

inhibi-tors; all Sigma) Cells were lysed by two passages through a

Stansted TC5W homogenizer at 12 000 lbÆin)2and kept on

ice throughout Insoluble debris was pelleted by

centrifuga-tion for 30 min at 48 400 g using a Beckman Avanti

J-20XP with JA-25.50 rotor, and the supernatant was

incu-bated by rotation for 1 h at 4C with 2 mL of amylose resin

Unbound protein was eluted by washing with 2· 10 mL of

buffer A, and bound protein was eluted after 15 min of

rota-ting incubation with 4 mL of elution buffer (buffer A with

10 mm maltose and 20% glycerol) The identity of proteins

of expected sizes for NBD1 and NBD2 were confirmed using

antibody to NBD1 [25] and an antibody to MBP (rabbit

polyclonal; New England Biolabs), respectively

Yields were typically  6 mgÆL)1 for NBD1–MBP and

0.8 mgÆL)1 for NBD2–MBP, and comprised > 95% of

total purified protein When not used fresh, purified

pro-teins were stored at) 80 C in 20% glycerol Proteins were

separated on 4–12% gradient Bis⁄ Tris gels and visualized

by Coomassie staining (Invitrogen)

MALDI-TOF MS MALDI-TOF MS (MS and MS⁄ MS) was performed using

a Bruker Ultraflex TOF⁄ TOF mass spectrometer (Bruker Daltonics, Coventry, UK) equipped with a nitrogen laser Gel bands of interest were digested in-gel according to standard procedures [26] using proteomics-grade Trypsin (Sigma-Aldrich) MS was performed using a-cyano-4-hy-droxycinnamic acid as matrix Peptide mass fingerprint spectra were matched against the NCBI nonredundant protein database using the search engine mascot (Matrix Science, London, UK) via an in-house license MS⁄ MS spectra were taken using the LIFT method (Bruker Dal-tonics) The accuracy of MS spectra was typically better than 50 p.p.m.: the accuracy of MS⁄ MS-generated frag-ment ions was in the range of ± 0.2 Da

Gel filtration Purified NBD–MBPs were diluted to 0.5 lgÆlL)1, and

300 lL was analyzed on a Superdex-200 gel filtration column (Tricon, Amersham Biosciences, Little Chalfont, UK) in AT-Pase buffer (50 mm Tris⁄ HCl, pH 7.2, 150 mm NH4Cl,

10 mm MgCl2) Relative protein size was calculated using Bio-Rad Gel Filtration standards (Cat no 151-1901) Peaks were collected and analyzed by EM, and for ATPase activity Purified SUR1F was mixed with DMPC⁄ DDM to final concentrations of 0.05% w⁄ v for DMPC and 0.1% w ⁄ v for DDM It was further concentrated to 1.45 mgÆmL)1, and

250 lL was loaded on a Superdex 200 (10⁄ 30) gel filtration column pre-equilibrated with 50 mL of buffer containing

50 mm Tris (pH 8.8), 150 mm NaCl, 0.05% w⁄ v DMPC and 0.1% w⁄ v DDM

EM and image processing For EM, protein samples were diluted to a concentration

of between 0.05 and 0.1 mgÆmL)1, applied to EM grids coa-ted with carbon film and stained with 2% uranyl acetate Preparations were examined using a CM120 electron micro-scope (FEI, Eindhoven, the Netherlands) with an acceler-ation voltage of 100 kV Electron micrographs were taken

at a magnification of · 45 000 Selected images were digit-ized with a step size of 25 lm on a Nikon Super Coolscan

9000 (Nikon, London, UK) The web and spider software packages [27] were used for all image processing In total,

524 particles were windowed, subjected to reference-free alignment, and sorted into 10 classes using the partitional method (K-means method) of clustering [28]

Nucleotide hydrolysis ATPase activity was normally measured for proteins puri-fied as above, but without the gel filtration step These

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