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Báo cáo khoa học: Reconstitution in vitro of the GDP-fucose biosynthetic pathways of Caenorhabditis elegans and Drosophila melanogaster ppt

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The assays were performed using GDP-Man as sub-strate; incubations were performed with extracts con-taining either of the Arabidopsis, Caenorhabditis or Drosophila enzymes alone or with

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pathways of Caenorhabditis elegans and Drosophila

melanogaster

Simone Rhomberg1, Christina Fuchsluger1, Dubravko Rendic´1, Katharina Paschinger1,

Verena Jantsch2, Paul Kosma1and Iain B H Wilson1

1 Department fu¨r Chemie, Universita¨t fu¨r Bodenkultur, Vienna, Austria

2 Abteilung fu¨r Chromosomenbiologie, Vienna Biocenter II, Austria

Fucose is a key component of many

oligosaccha-rides involved in recognition events and therefore

has roles in disease and development [1] For

instance, Notch, a protein involved in developmental

processes in animals, is modified with fucose

O-linked to the protein backbone [2], and a defect

in the relevant O-fucosyltransferase (POFUT1) is

lethal in mice [3], whereas the orthologous gene, nti,

is necessary for normal development of Drosophila

melanogaster [4,5] A second O-fucosyltransferase

(POFUT2) is also known, and RNAi in

Caenorhab-ditis elegans of the relevant gene, pad-2, results in severe body malformations [6] Less drastic are the effects of ablation of the FUT7 gene required for biosynthesis of certain fucose-containing selectin lig-ands; a lack of the encoded enzyme results in defi-cient leukocyte trafficking [7] On the other hand, certain fucosylated glycans are immunogenic and allergenic, an example of such a structure being the modification of the N-glycan core by a1,3-linked fucose [8] This feature is recognized by, e.g anti-(horseradish peroxidase), which is used to stain

Keywords

Caenorhabditis; Drosophila; GDP-fucose

biosynthesis; GDP-keto-6-deoxymannose 3,

5-epimerase ⁄ 4-reductase; GDP-mannose

dehydratase

Correspondence

I B H Wilson, Department fu¨r Chemie,

Universita¨t fu¨r Bodenkultur, Muthgasse 18,

A-1190 Vienna, Austria

Fax: +43 1 36006 6059

Tel: +43 1 36006 6541

E-mail: iain.wilson@boku.ac.at

Database

The nucleotide sequences of C elegans and

D melanogaster gmd and ger cDNA have

been submitted to the EMBL database

under accession numbers AM231683,

AM231684, AM231685, AM231686,

AM231687 and AM231688

(Received 30 November 2005, revised 17

February 2006, accepted 20 March 2006)

doi:10.1111/j.1742-4658.2006.05239.x

The deoxyhexose sugar fucose has an important fine-tuning role in regula-ting the functions of glycoconjugates in disease and development in mam-mals The two genetic model organisms Caenorhabditis elegans and Drosophila melanogaster also express a range of fucosylated glycans, and the nematode particularly has a number of novel forms For the synthesis

of such glycans, the formation of GDP-fucose, which is generated from GDP-mannose in three steps catalysed by two enzymes, is required By homology we have identified and cloned cDNAs encoding these two pro-teins, GDP-mannose dehydratase (GMD; EC 4.2.1.47) and GDP-keto-6-deoxymannose 3,5-epimerase⁄ 4-reductase (GER or FX protein; EC 1.1.1.271), from both Caenorhabditis and Drosophila Whereas the nema-tode has two genes encoding forms of GMD (gmd-1 and gmd-2) and one GER-encoding gene (ger-1), the insect has, like mammalian species, only one homologue of each (gmd and gmer) This compares to the presence of two forms of both enzymes in Arabidopsis thaliana All corresponding cDNAs from Caenorhabditis and Drosophila, as well as the previously uncharacterized Arabidopsis GER2, were separately expressed, and the encoded proteins found to have the predicted activity The biochemical characterization of these enzymes is complementary to strategies aimed at manipulating the expression of fucosylated glycans in these organisms

Abbreviations

GER, GDP-keto-6-deoxymannose 3,5-epimerase ⁄ 4-reductase; GMD, GDP-mannose dehydratase.

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neural tissue and cells in many invertebrates,

inclu-ding Caenorhabditis and Drosophila [9–11]

Not only have the relevant fucosyltransferases and

fucose-containing glycans been the object of study, but

also the proteins required for the biosynthesis and

transport of the fucose donor, GDP-Fuc, have been

examined This nucleotide sugar is generated de novo

from GDP-Man in three steps catalysed by two

cytoso-lic enzymes: GDP-mannose dehydratase (GMD; EC

4.2.1.47) and GDP-keto-6-deoxymannose

3,5-epi-merase⁄ 4-reductase (GER, otherwise known as

GDP-Fuc synthase; other synonyms include the FX protein

in man, the P35B tumour rejection antigen in mice, as

well as fcl or wcaG in bacteria and GMER in

Drosophila; EC 1.1.1.271) [12] In eukaryotes, GDP-Fuc

is then transported into the Golgi [13], the site of action

of, at least the majority of, the fucosyltransferases

To date, sequences encoding GMD and GER have

been cloned and expressed from man [14–16],

Arabid-opsis thaliana [17–19], Escherichia coli [20],

Helicobact-er pylori [21,22] and Paramecium bursaria Chlorella

virus 1 [23] Indeed Arabidopsis has two GMD genes,

one of which corresponds to the MUR1⁄ GMD2 gene,

a defect in which results in deficiencies in cell wall

bio-synthesis [24] GMD is also defective in the Chinese

hamster ovary Lec13 and murine lymphoma PLR1.3

mutant cell lines, and this absence results in resistance

to fucose-specific lectins [25,26] Mice defective in

GER suffer from postnatal failure to thrive and an

absence of leukocyte selectin ligand expression [27],

whereas mutant strains of both the intestinal symbiont

Bacteriodes and the nodulation symbiont

Sinorhizo-bium fredii unable to produce GDP-Fuc display

reduced colonization competitiveness in the presence

of wild-type strains [28,29] There also exists a

Dicytos-telium discoideum (slime mould) strain (HL250) with a

genetically undefined defect in the conversion of

GDP-Man into GDP-Fuc and a resultant reduced

germina-tion efficiency for older spores, suggesting that, as for

the aforementioned bacterial symbionts, the presence

of fucose may confer a selective advantage under

natural conditions [30] However, although early

stud-ies were taken to suggest that GMD may be defective

in patients with leukocyte adhesion deficiency II

(OMIM 266265) [31,32], it now appears to be accepted

that mutations in the GDP-Fuc transporter are the

reason for the observed reduction in fucosylation

[33,34] On the other hand, the high level of

fucosyla-tion in human hepatocellular carcinoma has been

cor-related, at least in part, with high expression of GER

and increased concentrations of GDP-fucose [35]

Considering that the enzymes involved in GDP-Fuc

biosynthesis in the two model invertebrates C elegans

and D melanogaster have not been studied to date, even though fucosylation appears to be important for their development [4–6], we sought to clone cDNAs predicted

to encode GMD and GER genes in these two organ-isms, using previously characterized A thaliana homo-logues as controls; indeed the encoded proteins were successfully expressed in bacteria and found, in concert,

to direct the synthesis of GDP-Fuc in vitro The two Drosophila enzymes GMD and GMER were also puri-fied; the GDP-Fuc product of these two enzymes was also characterized by NMR and by a functional assay

Results

Cloning and expression of Caenorhabditis and Drosophila GMD and GER cDNAs

Homologues of the human GMD protein were identified from Caenorhabditis and Drosophila, and the relevant cDNAs cloned Whereas Drosophila has, as previously determined in a theoretical study [36], one gmd gene (CG8890), Caenorhabditis has, like Arabidopsis [18], two gmd genes (gmd-1 and gmd-2 corresponding to the C53B4.7 and F56H6.5 Wormbase entries), which encode proteins that are 88% identical with each other (see Fig 1 for alignment) The Caenorhabditis gmd-1 gene is transcribed in two different forms resulting from use of different 5¢ exons (the second and smaller form, C53B4.7a, which is designated gmd-1a in this study); both 5¢-end gmd-1a EST clones in the databases contain

an SL1 spliced leader In the case of the second worm gene, encoding GMD-2, RT-PCR using a forward pri-mer containing the predicted start codon was unsuccess-ful, as was PCR using forward primers corresponding to the SL1 or SL2 spliced leaders and gmd-2-specific reverse primers Finally, gmd-2 was cloned in an incom-plete form starting with the second exon, which, how-ever, still contains the first region (Gly-Leu-Glu) conserved in comparison with the gmd-1 cDNAs

As for GMD, homologues of the human GER pro-tein were identified from Caenorhabditis and Droso-phila, and the relevant cDNAs cloned; we also cloned both Arabidopsis homologues The Drosophila homo-logue has already been named gmer (CG3495) [36], whereas the Caenorhabditis ger-1 corresponds to the R01H2.5 reading frame As for the GMD enzymes, alignments show a high degree of conservation between GER homologues (Fig 2)

Enzymatic activity of GMD and GER proteins All Arabidopsis, Caenorhabditis and Drosophila GMD and GER homologues were expressed using the

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pET30a system in the presence of kanamycin and

chlo-ramphenicol; in addition, a pCRT7-NT vector carrying

Caenorhabditis gmd-1 was also coexpressed with the

pET30a clone of Caenorhabditis ger-1 in the presence

of ampicillin, kanamycin and chloramphenicol Western blotting with an antibody to His showed for

Fig 1 Alignment of GMD sequences The following GMD protein sequences were aligned: C1 (C elegans GMD-1); C1a (C elegans GMD-1 alternatively spliced form, first 56 residues only); C2 (C elegans GMD-2); Dm (D melanogaster GMD); Hs (Homo sapiens GMD); Sj (Schisto-soma japonica GMD); Ec (E coli GMD); Pb (P bursaria Chlorella virus 1 GMD); A1 (A thaliana GMD1); A2 (A thaliana MUR1 ⁄ GMD2) Resi-dues conserved in comparison with the fly and worm sequences are highlighted, whereas key conserved resiResi-dues noted in the Discussion (GXXGXXG as well as the Ser ⁄ Thr residue and YXXXK motif catalytically important for SDR family members) are marked underneath with an asterisk.

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GMD homologues the expression of proteins of

 50 kDa, whereas in the case of the different forms of

GER the proteins were 35 kDa (Fig 3) The

Caenor-habditis and Drosophila GMD and GER proteins were tested for activity in coupled enzyme assays The Ara-bidopsis GMD2 (MUR1) and GER1 proteins were also

Fig 2 Alignment of GER sequences The following GER protein sequences were aligned: Ce (C elegans GER-1); Dm (D melanogaster GMER); Hs (Homo sapiens GER ⁄ FX); Sj (Schistosoma japonica GMD); Ec (E coli GMD ⁄ wcaG); Pb (P bursaria Chlorella virus 1 GER); A1 (A thaliana GER1); A2 (A thaliana GER2) Residues conserved in comparison with the fly and worm sequences are highlighted, whereas key conserved residues noted in the Discussion (GXXGXXG as well as the Ser ⁄ Thr residue and YXXXK motif catalytically important for SDR family members) are marked underneath with an asterisk.

Fig 3 Western blots of expressed GMD and GER isoforms GMD and GER proteins from A thaliana (MUR1, GMD1, GER1 and GER2),

C elegans (GMD-1, GMD-1a, GMD-2, GER-1 and coexpressed GMD-1 and GER-1) and D melanogaster (GMD and GMER) were expressed

in E coli BL21(DE3)pLysS cells for 2 h, and the soluble fractions of the bacterial proteins (equal lysate equivalents) were subjected to SDS ⁄ PAGE and western blotting using a primary antibody to His The sizes of the molecular mass standards are shown in kDa.

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tested as positive controls, as these have been

previ-ously shown to be enzymatically active when expressed

in E coli [17,19]

The assays were performed using GDP-Man as

sub-strate; incubations were performed with extracts

con-taining either of the Arabidopsis, Caenorhabditis or

Drosophila enzymes alone or with both enzymes from

the various species together The incubations were then

analysed by RP-HPLC, using authentic GDP-Man

and GDP-Fuc as external standards Initially, 0.5 m

KH2PO4was used as eluent [37], but, analogous to the

use of ammonium formate buffers for the purification

of UDP-xylose [38], it was then decided to examine the

use of the formate buffer As the results with the two

buffers were comparable, all subsequent analyses were

performed with the volatile formate buffer

Further-more, it was not absolutely necessary to perform the

GMD reaction before boiling and then adding GER;

such a procedure, though, has been described for

assays with E coli K12 Gmd and WcaG [39]

When GMD⁄ GER ‘pairs’ of any one of the three

species were present, a component that was coeluted

with standard GDP-Fuc was produced (Fig 4) In the

case of the Arabidopsis MUR1 and GER1 enzymes, the

putative GDP-Fuc product was shown to be a donor

substrate in fucosyltransferase assays (data not shown)

GDP-Fuc synthesis was also observed when either the

ArabidopsisMUR1 or the Caenorhabditis GMD-1a

iso-form were incubated with Caenorhabditis GER-1 and

GDP-Man (data not shown) In the absence of any

GER enzyme, but in the presence of any GMD, a

broad peak of intermediate retention time was

observed, as shown for Caenorhabditis GMD-1a and

Drosophila GMD (Fig 4A,D); this presumably

corres-ponds to the previously observed ketone and hydrate

forms of GDP-4-keto-6-deoxymannose [40] No

inter-mediate product was formed in the absence of any

GMD enzyme, and no GDP-Fuc was formed in the

absence of either GMD or GER, nor with the empty

vector control, showing that the strain of E coli used

has no detectable GDP-Fuc synthesis system The

chro-matograms also indicate that the amount of remaining

intermediate product was generally low or nonexistent

compared with the amount of GDP-Fuc, even though

GDP-Man was still present, and that the concentration

of GDP-4-keto-6-deoxymannose produced in the

pres-ence of GMD isoforms alone was greater than the

con-version of GDP-Man into products in the presence of

both enzymes (e.g with Drosophila GMD alone the

conversion of GDP-Man into the intermediate was

 80%, whereas in the presence of both enzymes the

conversion into GDP-Fuc was only  50%; compare

Fig 4A with 4C) This would indeed be compatible

with the feedback inhibition by GDP-Fuc previously shown for other forms of GMD also occurring to some extent with the fly and worm enzymes [14,39]

For the Caenorhabditis and Drosophila enzymes, expression at room temperature was necessary to detect activity: for the Drosophila enzymes, no activity was detected on expression at 37C, whereas for the Caenor-habditis enzymes, only minimal activity was found on expression at 16C GDP-Fuc synthesis on coexpres-sion of Caenorhabditis GMD-1 and GER-1 was margin-ally less efficient (12%) than synthesis in the presence of both separately expressed enzymes (15–20%) assayed under the same conditions; thus, there is no obvious requirement to coexpress GMD and GER This is unlike the situation with expression of the Arabidopsis MUR1

in yeast, as in this system MUR1 was susceptible to degradation when not coexpressed with GER1 [37]

Fig 4 Activity of expressed GMD and GER isoforms The soluble fractions of lysates (equal lysate equivalents) of bacteria expressing GMD and GER enzymes were incubated overnight with GDP-Man and subjected to RP-HPLC The chromatograms of the following combinations are shown: (A) Drosophila GMD alone; (B) Drosophila GMER alone; (C) Drosophila GMD and GMER; (D) Caenorhabditis GMD-1a; (E) Caenorhabditis GMD-1 and GER-1; (F) Caenorhabditis GMD-2 and GER-1; (G) Arabidopsis MUR1 (GMD2) and GER1; (H) Arabidopsis GMD1 and GER2 The elution positions of GDP-Man and GDP-Fuc standards are indicated In the experiments shown, the GMD and GER isoforms were expressed separately.

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In addition to using MUR1 and GER1 as controls,

we also examined the other Arabidopsis homologues

of these enzymes, respectively, GMD1 and GER2

Whereas GMD1 has been previously shown to be active

[18], GER2 was only identified in silico as a putative

epimerase-reductase [19] The assay data showed that,

as for MUR1 and GER1, incubations with both GMD1

and GER2 also resulted in synthesis of GDP-Fuc,

con-firming the activity of GER2 for the first time (Fig 4H)

Properties of GMD and GER proteins from

different species

Initially, the composition of the assay mixture used was

based on previously published procedures [14]

There-fore, to test the limits of the system, we performed

assays with the Arabidopsis enzymes in the absence of

one of each of the nonenzymatic components

Compar-able to previous reports, we found that the synthesis by

GER1 of GDP-Fuc from the intermediate product was

absolutely dependent on NADPH, whereas reducing

the NADPH concentration by half, or increasing it

twofold, had no influence on the yield of GDP-Fuc

Furthermore, reduced conversion of the intermediate

was observed in the absence of dithiothreitol On the

basis of these data, we did not alter the assay mixture

composition for the later assays However, to optimize

the preparation of GDP-Fuc in a ‘one-pot’ method, we

also examined the pH and temperature requirements

for its production from GDP-Man

The Drosophila enzymes, taken together, displayed a

relatively broad pH optimum (pH 5–8), resulting,

under the conditions used, in 50% conversion of

GDP-Man into GDP-Fuc Caenorhabditis GMD-1 and

GER showed, in combination, optimal activity at

pH 8–9 (15–20% conversion using the same amount

of soluble bacterial extract as for the Drosophila

enzymes); similarly, an optimum at pH 8 was reported

for the synthesis of GDP-Fuc by Aerobacter aerogenes

and CHO cell extracts [25,41], whereas both the

sepa-rately assayed GMD and GER from porcine thyroid

show optima at pH 7 [42,43] and recombinant forms

of human and E coli GMD have optima of pH 7.5–

8.0 [32,44] The recombinant E coli K-12 GER

enco-ded by the wcaG gene was most active in the range

pH 6–7 [45]

As regards temperature, the Drosophila enzymes

were most active at temperatures of 16–30C, whereas

the Caenorhabditis enzymes (specifically GMD-1 and

GER-1) displayed a temperature optimum of 23–37C

(Fig 5) Assays with recombinant GDP-mannose

dehydratases alone showed that both Caenorhabditis

GMD-1a and Drosophila GMD had temperature

optima  30 C, whereas Caenorhabditis GMD-2 was most active at 16–23C (data not shown)

Purification of Drosophila GMD and GMER

In the preceding studies, the identity of the GDP-Fuc product was based on HPLC retention time; thus, it was decided to purify the product of the fruitfly pro-teins for further analysis Thus Drosophila GMD and GMER were subjected to nickel-chelation chromato-graphy either separately or together and isolated after elution with 250 mm imidazole (Fig 6, upper panel) The dominant bands (35 kDa and 50 kDa, corres-ponding to GMER and GMD, respectively) eluted with the latter buffer reacted with an antibody to His (Fig 6, lower panel), and their identity was verified by MALDI-TOF tryptic peptide mapping Protein assays indicated that the yields of individually purified Drosophila GMD and GMER were  0.5 mg from a 50-mL culture; when purified together, the total pro-tein yield was 1 mg Under the conditions used, the yield of GDP-Fuc with the separately purified enzymes was comparable to that using enzymes purified together; it also appeared that, after purification, GMER was more stable than GMD (data not shown) Using the purified forms of Drosophila GMD and GMER, scaled-up incubations were performed, prepu-rified by passage over a small Lichroprep column and subjected to RP-HPLC to yield an estimated total of

 1 mg GDP-Fuc (20% yield after purification) This material was lyophilized and used successfully as a fuc-osyltransferase substrate when an extract of Sf9 cells transfected with Drosophila core fucosyltransferase

120 100 80 60 40 20 0

Ara Ce Dm

Temperature [ºC]

Fig 5 Relative yield of GDP-Fuc with respect to incubation tem-perature Assays of Arabidopsis MUR1 and GER1, Drosophila GMD and GMER, and Caenorhabditis GMD-1 and GER-1 were performed

at different temperatures, and the relevant RP-HPLC peaks were integrated The data were then recalculated individually for each enzyme pair relative to the respective activity at 23 C.

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FucTA was used as an enzyme source as judged by the

conversion of the dabsyl-GnGnF6 glycopeptide

sub-strate into a species with an m⁄ z 146 higher (data not

shown) Furthermore, the compound was subjected to

NMR, which confirmed its identity as GDP-Fuc (Table 1), the data matching those reported for syn-thetic GDP-Fuc [46]

Developmental expression profile in Caenorhabditis

Considering the multiplicity of genes and transcripts encoding GDP-mannose dehydratase in C elegans, semi-normalized RT-PCR was performed using cDNA from L1 larvae, L2⁄ 3 larvae (combined as these are difficult to distinguish), L4 larvae and adults The results (Fig 7) would suggest minor variations in the concentrations of the gmd-2 and ger-1 transcripts dur-ing worm development A peak of gmd-1 transcription may be occurring in the L2⁄ 3 stage, but transcripts of this form are seemingly under-represented in adults

Fig 6 Purification of recombinant Drosophila GMD and GMER.

GMD and GMER expressed separately were subjected to nickel

chelation chromatography; fractions marked ‘co’ are from the

purif-ication of GMD and GMER from mixed lysates Fractions (wash,

20 m M imidazole and 250 m M imidazole) were then

electrophor-esed and stained using Coomassie blue (upper panel) or transferred

to nitrocellulose and probed with an antibody to His (lower panel).

In the case of copurification, some GMER, but no GMD, was

elut-ed with 20 m M imidazole (lane 4) The sizes of the molecular mass

standards are shown in kDa.

Table 1 NMR data of GDP-b- L -fucose prepared using recombinant Drosophila enzymes ND, not determined Further signals at 3.76 p.p.m.

in the proton spectrum and at 60.16 in the carbon spectrum are from residual Tris buffer.

J (Hz) J PP 20.5, J HP 8.0

13

Fig 7 Development RT-PCR profile for GMD and GER transcripts

in Caenorhabditis RT-PCR was performed using RNA isolated from L1, L2 ⁄ L3, L4 and adult C elegans using primers specific for gmd-1, gmd-1a (alternatively spliced form of GMD-1), gmd-2 and ger-1 The amounts of cDNA used in the PCRs were normalized on the basis of the intensity of actin transcripts.

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On the other hand, the alternatively spliced gmd-1a

transcript is present at its lowest concentrations in L1

larvae and is relatively more abundant in the later

sta-ges The expression of the GDP-Fuc biosynthesizing

enzymes throughout development is compatible with

the rich variety of fucosylated N-glycans and

O-gly-cans in this species [47,48]

Discussion

GDP-fucose was first found in 1958 [49], and its

bio-synthesis is a process present in many life forms, from

bacteria through to plants, invertebrates and

verte-brates There appear to be two basic strategies for the

formation of GDP-Fuc: either the route through

GDP-Man, using GMD and GER enzymes, or the

‘salvage’ pathway through fucose 1-phosphate The

first route was first found in A aerogenes and shown

to be dependent on the presence of NADPH (then

called TPNH) [50] GMD was first isolated from

Phaseolus vulgaris [51], whereas GER was initially

purified from porcine thyroid [43] The presence of a

fucose salvage pathway was first suggested in 1964

because of the ability to radiolabel glycoproteins after

administration of [14C]Fuc to rats [52] and confirmed

by detection of l-fucose kinase and GDP-l-Fuc

pyro-phosphorylase [53,54] in porcine liver More recent

studies indicate that there are varying levels of

fuco-kinase activity in different rat tissues [55]

Some organisms have both pathways, as shown by

biochemical work; initially both routes were

consid-ered to be only present in mammals, but now the

two pathways have been demonstrated in Bacteriodes

[28] The genomic ‘revolution’, however, means that

further phylogenetic analyses can now be performed

By this approach, it can be seen that the

GMD⁄ GER route is probably present in all

organ-isms known to produce fucose-containing

glycoconju-gates; on the other hand, as noted previously,

Drosophila has no genetically detectable ‘salvage’

pathway Plants and mammals do have relevant

homologues, although the putative plant ‘salvage’

pathway is seemingly closer to that of Bacteriodes,

as plant genomes contain homologues of the fkp

gene from Bacteriodes, a gene that encodes a protein

with both fucokinase and GDP-Fuc phosphorylase

activities [28] In mammals, however, these activities

are encoded by separate genes Caenorhabditis

appears, on the other hand, only to have an obvious

fucokinase homologue (C26D10.4) In addition,

GMD is also required for the de novo synthesis of

GDP-Rha in Ps aeruginosa, as the product of

GMD, GDP-4-keto-6-deoxy-d-mannose, can also be

acted on by a reductase [56], whereas the GMD of the P bursaria Chlorella virus 1 can directly convert GDP-4-keto-6-deoxy-d-mannose into GDP-Rha [23] Thus it is conceivable that GMD is more ancient than GER

The GMD and GER sequences across the various kingdoms of life are remarkably highly conserved; both proteins are members of the short chain dehy-drogenase (SDR) family and display homologies to other enzymes of sugar nucleotide metabolism, such as dTDP-glucose dehydrogenase, UDP-Gal epimerase and UDP-GlcA decarboxylase Phylogenetic trees (not shown) suggest that the plant enzymes are closer to the bacterial, than to the animal, ones; regardless of this, however, residues found by crystallographic or mutagenesis studies to be important for binding or catalysis are identical across all sequences A Ross-mann motif (Gly-Xaa-Xaa-Gly-Xaa-Xaa-Gly), which

is a common feature of nucleotide-binding proteins, is, for instance, conserved in all the GMD and GER sequences from worm and fly Furthermore, the resi-dues corresponding to Gln39, Asp40, Ser117 and Arg220 in MUR1 3D structure, which form hydrogen bonds with the NADPH cofactor, and the residues that correspond to Asn214, Lys228, Arg253, Arg314 and Glu317 of the MUR1 sequence and form hydro-gen bonds with the GDP moiety [57] are retained in the worm and fly GMD enzymes The Ser⁄ Thr residue and Tyr-Xaa-Xaa-Xaa-Lys motif catalytically import-ant for SDR family members are also conserved in all sequences If the corresponding Ser⁄ Thr, Tyr and Lys residues of the E coli GMD or GER are separately subjected to site-directed mutagenesis, then either activity is abolished or the kcat drastically decreased [58,59]

It is also noteworthy that some organisms have mul-tiple GMD or GER genes In particular, Arabidopsis has two proven GMD enzymes (GMD1 and MUR1; At5g66280 and At3g51160), displaying differential expression [18], as well as the previously proven GER1 and now, by us, proven GER2 (respectively, At1g73250 and At1g17890): in both cases the genes are in, or at least close to, regions that have putatively been duplica-ted during the evolution of Arabidopsis [60,61] Further-more, the presence of duplicated genes means that knocking-out one GMD, i.e MUR1, does not totally diminish the fucose content of Arabidopsis glycoconju-gates [24] Any strategy to abolish all fucosylation in plants is possibly also complicated by the presence of the aforementioned fkp homologue On the other hand, Drosophila has only one GMD and one GER homo-logue; indeed, a GMD mutation has been isolated and

is lethal at the third larval stage [62], commensurate

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with the putative key role for peptide

O-fucosyl-transferases in development and the probable lack of

any salvage pathway

C elegans, however, is somewhere between these

extremes, as it has two GMD enzymes (although the

related nematode Caenorhabditis briggsae appears to

have only one gmd gene, suggesting that the duplication

of gmd genes is an evolutionarily relatively recent event),

whose activities were proven in the course of our studies,

but only one GER isoform Suggestive of functional

degeneracy are RNAi data on the two Caenorhabditis

GMD homologues: at least when performed

individu-ally, as part of a large-scale screen, RNAi of gmd-1,

gmd-2and ger-1 resulted in no obvious associated

lethal-ity However, in another large-scale RNAi screen with

the hypersensitive rrf-3 worm strain, various defects

were indeed reported upon knock-down of gmd-2 [63];

no data, however, on gmd-1 or gmd-1a were reported in

the study using rrf-3 worms, so neither the relative

importance of the two genes nor the biological

signifi-cance of the alternative splicing of gmd-1 can be

sur-mised at present Owing to the previous report as to the

effect of mutations in the C elegans POFUT2

homo-logue pad-2 [6], the Drosophila orthohomo-logue of which

modifies thrombospondin repeats [64], it is quite

prob-able that any effects of RNAi targeting of GDP-Fuc

biosynthesizing enzymes will be due to peptide

O-fuco-sylation defects There may also be tissue-specific

expression of the two gmd genes Apparently, gmd-1 is

expressed in body wall muscle and head neurons (For

summaries of the various RNAi and expression data,

see: http://www.wormbase.org/db/gene/gene?name¼

C53B4.7 for gmd-1, http://www.wormbase.org/db/gene/ gene?name¼ F56H6.5 for gmd-2 and http:// www.wormbase.org/db/gene/gene?name ¼ R01H2.5 for ger-1.) Our own developmental RT-PCR profile data would suggest that there is no major developmental regulation of the transcription of either GMD-encoding gene, although there appears to be a peak of gmd-1 and gmd-1a expression at the L2⁄ L3 stage; such results, however, are not incompatible with variation of expres-sion within tissues and may reflect a requirement for higher concentrations of GDP-Fuc at certain times or

in certain tissues during development (e.g for Notch signaling)

In summary, we have shown for the first time that the GMD and GER homologues of Caenorhabditis and Drosophila, as well as the Arabidopsis GER2, are indeed functional enzymes, which can work together to reconstitute GDP-Fuc synthesis in vitro Biochemical characterization of these enzymes lends confidence to any subsequent reverse genetic or phylogenetic studies

or in the use of conditional mutants and lays the foun-dation for future work on the role of fucose in the biology of these model organisms

Experimental procedures

Cloning of GMD and GER cDNAs

RNA was extracted from A thaliana (Columbia), C elegans (N2) or D melanogaster (Canton S) using Trizol reagent (Invitrogen, Paisley, UK) Two-step RT-PCR was performed using Superscript III reverse transcriptase (Invitrogen) and

Table 2 Primers used in this study.

AtGMD1 ⁄ 2 ⁄ EcoRI, CGGAATTCAAGGTCGTGCTGAGCTC (rev)

AtMUR1⁄ 2 ⁄ XhoI, ACCCTCGAGTCAAGGTTGCTGCTTAGC (rev)

AtGER1⁄ 2 ⁄ XhoI, ACCCTCGAGTTATCGGTTGCAAACATTCTT (rev)

AtGER2⁄ 2 ⁄ XhoI, CCGCTCGAGTTACTGCTTCTTCTGCACAA (rev)

CeGMD ⁄ 1 ⁄ BamHI, CGGGATCCAATGCCAACCGGCAAGTCTG (fwd),

or CeGMD1a ⁄ 1 ⁄ NcoI, CATGCCATGGCTGATCAAAATGCGAA (fwd) CeGMD1⁄ 2 ⁄ HindIII, CCCAAGCTTAAGCCATTGGATTGGACTTC (rev)

CeGMD2⁄ 1 ⁄ BamHI, CGGGATCCTAAGCCATTGGATCTGCC (rev)

CeGER ⁄ 2 ⁄ EcoRI, CGGAATTCTTATTTTCTAGCCGTCTCATAA (rev)

DmGMD⁄ 2 ⁄ XhoI, CCGCTCGAGTTAAGCGATTGGATTTTTCCT (rev)

DmGMER ⁄ 2 ⁄ XhoI, CCGCTCGAGTTACTTTCTAGCCTGGTCG (rev)

Trang 10

Expand polymerase (Roche, Vienna, Austria) using the

pri-mer pairs listed in Table 2 The PCR fragments were cut and

ligated into either pET30a (Novagen, Merck Biosciences,

Darmstadt, Germany) or pCRT7-NT (Invitrogen) digested

with the relevant restriction enzyme(s) DNA sequencing was

performed using the BigDye kit (Applera, Norwalk, CT,

USA) In the case of the Caenorhabditis gmd-1 and ger-1 pET

clones, the second codon encodes alanine (respectively a

replacement codon or an additional one) in order to

accom-modate an NcoI site

Expression of GMD and GER proteins

Plasmids were used to transform BL21(DE3)pLysS Gold

cells (Stratagene, Amsterdam, the Netherlands), which were

grown overnight in 10 mL Luria–Bertani medium containing

kanamycin and chloramphenicol (also containing ampicillin

in the case of double transformation) In the case of trial

expression, 1 mL (or 2.5 mL for larger-scale cultures) was

taken from the overnight culture to inoculate 20 mL (or

50 mL) Luria–Bertani medium containing the relevant

anti-biotics After the A600had reached 0.6 at 37 C, small-scale

cultures were split, and to one half was added isopropyl

b-d-thiogalactoside to a concentration of 1 mm; in the case

of larger-scale cultures, isopropyl b-d-thiogalactoside was

added to the entire culture The growth was continued at

23C for up to three hours

Cells were resuspended in 500 lL (small-scale) or 5 mL

(large-scale) lysis buffer containing 50 mm Tris, 400 mm

NaCl, 100 mm KCl, 10% glycerol, 0.5% Triton X-100,

10 mm imidazole, pH 7.8, and lysed by performing repeated

freeze–thaw cycles, using alternately a methanol bath and a

42C water bath DNase I was added, and the lysates were

incubated for 10 min at 37C before centrifugation for

1 min (small-scale) or 20 min (large-scale) at 14 000 g,

4C The supernatant was taken for assays or, in the case

of large-scale cultures, purification For the presented data,

the cells were always grown and lysed under the same

con-ditions (i.e same initial cell density, temperature, time of

induction and concentration of isopropyl

b-d-thiogalacto-side) Aliquots of these lysates stored at )80 C still

dis-played activity after 1 year of storage

Purification by nickel-chelation chromatography

The supernatants from lysed cells were incubated with 2 mL

Ni⁄ nitrilotriacetate resin (Qiagen, Vienna, Austria) for at

least 1 h at 4C The lysate ⁄ resin mixture was poured into a

column at room temperature and washed twice with 1 mL

lysis buffer, before further washing twice with 4-mL aliquots

of a lysis buffer containing 20 mm imidazole Elution was

performed using four 0.5-mL aliquots of a lysis buffer

con-taining 250 mm imidazole All fractions were collected on ice

Protein assays were performed using the modified Lowry kit

(Sigma, Vienna, Austria)

Western blotting

Aliquots of the soluble fractions of lysed bacteria or of affinity chromatography fractions (20 lL) were precipitated with a fivefold excess of cold methanol and, after 1 h at )20 C, centrifuged (14 000 g, 5 min) After removal of residual methanol at 65C, the samples were resuspended

in Laemmli sample buffer (20 lL) and denatured at 95C for 5 min; 5 lL of these samples were subjected to SDS⁄ PAGE with subsequent blotting on to nitrocellulose Recombinant His-tagged forms of GMD and GER were then detected using antibody to His (HIS-1; Sigma; 1 : 3000 dilution) followed by anti-mouse IgG (Fc or c-specific) con-jugated with alkaline phosphatase (Sigma; 1 : 10 000 dilu-tion) and use of SigmaFAST BCIP⁄ NBT substrate

Assay of GMD and GER activity

To determine the enzymatic activity, aliquots of crude sup-ernatants of lysed cells or of purified proteins (2 lL) were typically incubated at room temperature in the presence

of 20 mm Tris⁄ 5 mm EDTA ⁄ 10 mm dithiothreitol ⁄ 1 mm GDP-mannose⁄ 5 mm NADPH ⁄ 1 mm NAD+

, pH 7.4 (final volume 10 lL) RP-HPLC was then performed using a Hypersil column with isocratic elution using 600 mm ammonium formate, pH 3.2 Peak integrations were used

to estimate the yield of either GDP-4-keto-6-deoxymannose (GMD assays) or GDP-Fuc (combined GMD⁄ GER assays)

NMR analysis

Approximately 1 mg of the HPLC-purified reaction product

of GDP-Man with purified Drosophila GMD and GMER was lyophilized twice and taken up in D2O before NMR analysis Spectra were recorded at 300 K at 300.13 MHz for1H, at 75.47 MHz for 13C, and at 121.49 MHz for 31P with a Bruker AVANCE 300 spectrometer equipped with a 5-mm QNP-probehead with z gradients Data acquisition and processing were performed with the standard xwinnmr software (Bruker BioSpin GmbH, Rheinstetten, Germany)

1

H-NMR spectra were referenced to 2,2-dimethyl-2-silapen-tane-5-sulfonic acid (d¼ 0), 13

C-NMR spectra were refer-enced externally to 1,4-dioxane (d¼ 67.40), and 31P-NMR spectra were referenced externally to H3PO4 (d¼ 0) HMQC and HMBC spectra were recorded in the phase-sensitive mode using TPPI and pulsed field gradients for coherence selection

Developmental transcript analysis

A total of 120 individual Caenorhabditis L1 larvae and

60 individuals of three other stages (L2⁄ L3 larvae, L4 larvae and adults) were picked; the RNA, extracted using

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