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The evolution of the structure and function of the transthyretin homolog [referred to as transthyretin-like protein TLP] has been the focus of recent studies by several research groups..

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Evolutionary changes to transthyretin: structure and

function of a transthyretin-like ancestral protein

Sarah C Hennebry

Department of Biochemistry and Molecular Biology, Bio21 Institute, The University of Melbourne, Victoria, Australia

Introduction

The evolution of the structure and the function of the

thyroid hormone (TH) distributor, transthyretin, has

been well researched The primary, secondary, tertiary

and quaternary structures of this vertebrate protein are

highly conserved It was therefore hypothesized that

the transthyretin gene may have evolved in a

nonverte-brate organism Searches for a transthyretin progenitor

led to the identification of a transthyretin homolog,

which was found initially in nonvertebrate genomes and subsequently in all major kingdoms The evolution

of the structure and function of the transthyretin homolog [referred to as transthyretin-like protein (TLP)] has been the focus of recent studies by several research groups TLPs from various organisms have been demonstrated to share remarkable structural similarities to vertebrate transthyretins Despite this

Keywords

evolution; purines; structure; transthyretin;

transthyretin-like protein

Correspondence

S C Hennebry, Human Neurotransmitters

Laboratory, Baker IDI Heart and Diabetes

Institute, P.O Box 6492, St Kilda Road

Central Melbourne, Victoria 3008, Australia

Fax: +61 3 8532 1100

Tel: +61 3 8532 1734

E-mail: sarah.hennebry@bakeridi.edu.au

(Received 2 February 2009, revised 8 June

2009, accepted 8 July 2009)

doi:10.1111/j.1742-4658.2009.07246.x

The structure of the thyroid hormone distributor protein, transthyretin, has been highly conserved during the evolution of vertebrates Over the last decade, studies into the evolution of transthyretin have revealed the exis-tence of a transthyretin homolog, transthyretin-like protein, in all king-doms Phylogenetic studies have suggested that the transthyretin gene in fact arose as a result of a duplication of the transthyretin-like protein gene

in early protochordate evolution Structural studies of transthyretin-like proteins from various organisms have revealed the remarkable conservation

of the transthyretin-like protein⁄ transthyretin fold The only significant differences between the structures of transthyretin-like protein and transthyretin were localized to the dimer–dimer interface and indicated that thyroid hormones could not be bound by transthyretin-like protein All transthyretin-like proteins studied to date have been demonstrated to function in purine metabolism by hydrolysing the oxidative product of uric acid, 5-hydroxyisourate The residues characterizing the catalytic site in transthyretin-like proteins are 100% conserved in all transthyretin-like protein sequences but are absent in transthyretins Therefore, it was proposed that following duplication of the transthyretin-like protein gene, loss of these catalytic residues resulted in the formation of a deep, negatively charged channel that runs through the centre of the transthy-retin tetramer The results thus demonstrate the remarkable evolution of the transthyretin-like protein⁄ transthyretin protein from a hydrolytic enzyme to a thyroid hormone distributor protein

Abbreviations

5-HIU, 5-hydroxyisourate; COG, cluster of orthologous groups; OHCU, hydroxy-4-carboxy-5-ureidoimidazoline; PTS2, type-two peroxisomal sequence; RNAi, RNA interference; TH, thyroid hormone; TLP, transthyretin-like protein.

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structural similarity, TLP and transthyretin have

dif-ferent functions TLP is an enzyme functioning in the

purine catabolism pathway, where it hydrolyses

5-hy-droxyisourate (5-HIU), the oxidation product of uric

acid Phylogenetic analyses have revealed that it is

likely that the transthyretin gene arose as a result of a

duplication of the TLP gene in early vertebrate

evolu-tion Thus, the evolution of TLP and transthyretin

rep-resents a remarkable case of the divergent evolution

from an enzyme to a hormone distributor

This minireview will present and discuss recent

find-ings regarding the identification and distribution of

TLP genes in nature, the structural and functional

characterization of the TLP from various organisms,

and the evolution of TLP and transthyretin

The identification of TLPs and

transthyretins in nature

The evolution of transthyretin and its distribution in

nature have been well researched [1,2] Several studies

have demonstrated that all vertebrates synthesize

transthyretin at some stage during their development

[2–4] and this synthesis is primarily localized to the

liver, choroid plexus and retinal pigment epithelium

The expression of the transthyretin gene in vertebrates

occurs independently in these tissues [5,6] There is

considerable sequence identity and similarity between

the amino acid sequences of transthyretin from various

vertebrate organisms The most divergent transthyretin

sequences (for example, human and sea bream

trans-thyretin) still retain 67% similarity (48% identity)

This consensus of primary structure is also reflected in

the highly conserved secondary, tertiary and

quater-nary structures of transthyretin from vertebrates

Together, these data suggest that the transthyretin

gene may have evolved before the divergence of

verte-brates from inverteverte-brates

The genomics era has been characterized by the

increasingly rapid sequencing of multiple genomes

alongside the development of sophisticated pairwise

sequence-alignment search tools These were the

cata-lysts enabling the search for a transthyretin homolog

among nonvertebrate organisms The first evidence

for such a homolog was published in 2000 [7], when

blastsearches [8] revealed the existence of ORFs with

the potential to encode a protein of similar length

and sequence composition to transthyretin These

ORFs were initially identified in the enteric bacteria

Escherichia coli and Salmonella dublin, in the yeast

Schizosaccharomyces pombe and in the nematode

Caenorhabditis elegans[7] The predicted protein

homo-log of transthyretin was termed TLP because the name

transthyretin implied a role in the transport of thyroid hormones and retinol binding protein [9] Such a func-tion could not be assumed for the nonvertebrate trans-thyretin homolog

Sequence characteristics of TLP and transthyretin

With the availability of an increasing number of genomes to mine, Eneqvist et al [10] used blast searches

to identify a further 49 putative TLP sequences in the genomes of bacteria, plants and invertebrate animals The TLP genes they identified typically encoded a pro-tein of 114 amino acid residues compared with, on average, 125 residues in transthyretin (the number of residues was species dependent) Furthermore, Eneq-vist et al [10] observed that all TLP sequences possessed a consensus C-terminal tetrapeptide: Tyr-Arg-Gly-Ser Alignment of TLP and transthyretin sequences revealed that the regions of greatest similar-ity between the two families of proteins were in the N-terminal and C-terminal regions [11] In order to distinguish between the two protein families in greater detail, a comparative analysis of TLP and transthy-retin sequences was performed [11] In this study, a set

of bacterial TLP and vertebrate transthyretin sequences was probed for motifs that might be con-served in each group The study revealed that the transthyretin sequences in this set were so similar that

a single motif spanned the entire length of each protein sequence However, in the set of TLP sequences, five specific motifs were identified, namely motifs A–E (with motif A being the most highly conserved) The motifs in the TLP sequences were found in the follow-ing arrangement (from N-terminal to C-terminal): (E)-B-D-C-A (see Fig 1A) Motif E was only found in TLPs from plant species and from two alphaproteo-bacteria: Bradyrhizobium japonicum and Magnetospiril-lum magnetotacticum Motif E is homologous to the proteins of cluster of orthologous groups (COG)

3195, a group of bacterial proteins where the entire protein is made up of this single domain Motif E has been subsequently identified as a unique protein, 2-oxo-4-hydroxy-4-carboxy-5-ureidoimadolazine (OHCU) decarboxylase, whose function relative to TLP will be discussed later in this review

A combined set of TLP and transthyretin sequences was also probed for motifs to determine whether there were any motifs in common between the two protein families Three motifs (A’–C’), which highlighted regions of similarity between TLP and transthyretin sequences, were identified and found in the arrange-ment B’-C’-A’ (see Fig 1B) These motifs were shown

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to correspond to regions of structural significance in

the transthyretin molecule (see Fig 1C) Motif A’

cor-responds to residues that line the hydrophobic core of

the transthyretin tetramer Motif B’ corresponds to

residues forming the dimer–dimer interface and

resi-dues in motif C’ are involved in monomer–monomer

interactions (see Fig 1C) Based on these observations,

it was hypothesized that TLP probably has a tertiary

structure similar to that of transthyretin [11] The

motifs identified in this study also provided a more

accurate means of differentiating between TLP and

transthyretin sequences and for the identification of

novel TLP⁄ transthyretin sequences through Hidden

Markov searches in protein databases [11]

Interestingly, whilst motifs A’–C’ represent the

regions of greatest sequence similarity between TLP

and transthyretin, they also contain specific amino acid

substitutions that enabled the distinction of one group

from the other For instance, at their C-termini (motif

A’ region), nearly all TLP sequences possess a

Tyr-Arg-Gly-Ser tetrapeptide Specifically, the tyrosine and

glycine residues were found to be 100% conserved

among TLP sequences Upon sequence alignment with

TLP, the residues at the same positions in transthyretin are threonine and valine, respectively At the N-termini

of TLP sequences (the motif B’ region) a conserved his-tidine residue was found The equivalent residue in transthyretin sequences is lysine (also 100% conserved) Interestingly, the residues involved in TH binding in transthyretin are not conserved in TLP sequences Rather, it appears that residues involved in the struc-tural integrity of the TLP⁄ transthyretin molecule have been conserved The alignment of representative trans-thyretin and TLP sequences in Fig 2 demonstrates the distribution of residues that are 100% conserved in both TLP and transthyretin sequences as well as those that are 100% conserved solely within the set of TLP sequences

Distribution of TLPs and transthyretins

in nature

The distribution of TLP in nature and its evolutionary relationship to transthyretin have been studied exten-sively in recent years [10,11] To date, TLP genes have been identified in over 200 organisms across all

king-A

B

C

Motif A

A

A C D B

C

~ 127 amino acids

~ 114 amino acids

B

Motif B

Motif C

Fig 1 Motifs common between TLP and

transthyretin indicate conservation of the

TLP ⁄ TTR structure through evolution Motifs

identified in (A) TLP sequences and (B)

transthyretin+TLP sequences (A) In the set

of TLP sequences, four motifs were

identi-fied (A–D) The motifs are found in the order

B-D-C-A, with A being the most highly

con-served (B) In the set of transthyretin+TLP

sequences, three motifs were identified,

A’–C’ Motif A’ is equivalent to motif A from

the TLP motif set Motif B’ is similar but

extended in the N-terminal and C-terminal

regions to motif B Motif C’ is shorter than

motif C and its location is shifted towards

the N-terminus Motif D is specific to the

TLP set of proteins (C) Motifs A’–C’ were

superimposed on the tertiary structure of

sea bream transthyretin Motif A’ lines the

hydrophobic core Motif B’ forms the

dimer–dimer interface and the opening of

the central channel of the TTR molecule.

Residues in motif C’ are involved in

mono-mer–monomer interactions (Modified from

[11]).

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doms By contrast, the transthyretin gene is only found

in vertebrates Whilst the TLP gene is widely distributed

in nature, there are some notable absences or apparent

‘losses’ of the TLP gene For instance, no protozoans to

date have been found to have a TLP gene, even though

related organisms such as the slime mold

Dictyosteli-um discoideDictyosteli-um and the jakobite Jakoba bahemiensis

both express the TLP gene A TLP gene is absent from

the cnidarian and ascidian phyla, despite the fact that

organisms before and after these branch points in

evo-lution express the TLP gene This evidence suggests that

whilst TLP might have been conserved throughout

evo-lution because they have an important functional role,

it is by no means essential to all organisms

Subcellular localization of TLP in

bacteria

In most instances, the TLP gene is present as a single

copy in the organisms in which it has been identified

The gene typically encodes a cytoplasmic protein and,

in the case of bacteria, is typically located in purine

metabolism operons, neighbouring the gene which

encodes OHCU decarboxylase [11] This is consistent

with the recently determined role of TLP in this

meta-bolic pathway (to be discussed later) A notable

excep-tion to this is the case of the enterobacterial TLP genes and a handful of TLP genes from other Gram-negative bacteria The TLPs from these bacteria have been found to possess an N-terminal extension, namely

a periplasmic localization sequence [11] Interestingly, these TLP genes are not found to be associated with purine metabolism operons [11], and it is therefore tempting to speculate that their primary function is not purine metabolism

Some organisms have multiple copies of a TLP gene (see Table 1) [11] In these cases, one gene encodes a cytoplasmic TLP and the ‘additional’ TLP gene encodes a periplasmic protein that, similarly to entero-bacterial TLP genes, is not associated on the entero-bacterial chromosome with genes encoding proteins involved in purine metabolism Indeed, phylogenetic analyses of all periplasmic TLP sequences (S.C Hennebry, unpub-lished results) suggests that the genes encoding these TLPs were probably obtained through horizontal gene transfer from an enterobacterial ancestor

Subcellular localization of TLPs in eukaryotes

In most nonfungal eukaryotic TLP sequences exam-ined to date, an N-terminal extension has also been

Fig 2 Alignment of representative transthyretin and TLP sequences Mature amino acid sequences for transthyretin and TLP from selected organisms are shown (i.e with signal peptides removed) The shared secondary structure characteristics of transthyretins and TLPs are indi-cated above the alignment: motifs A’–C’ are indiindi-cated with straight lines and are labelled; b-strands are indiindi-cated with arrows and are labelled A–H A single a-helix is indicated with a rectangle The residues that are strongly conserved between transthyretins and TLPs are indicated with an asterisk (*) Residues 100% conserved among all TLP sequences are indicated with a hash (#) Numbering for human transthyretin is shown directly beneath the alignment.

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identified This N-terminal extension contains a

nona-peptide, which is predicted to encode a type-two

peroxisomal sequence (PTS2) [11,12] Recently, a

proteomic analysis of leaf peroxisomes confirmed the

peroxisomal localization of the Arabidopsis thaliana

TLP [13] Found in all eukaryotic cells, peroxisomes

are specialized organelles in which oxidative reactions,

such as those associated with purine metabolism, are

compartmentalized The co-localization of

purine-metabolism enzymes (e.g uricase) with TLP in

peroxi-somes is therefore in keeping with the function of the

A thalianaTLP hydrolysis of the purine 5-HIU (S C

Hennebry, unpublished results) These observations

contradict those made by Nam and Li [14], where the

A thaliana TLP was reported to be localized only in

the cytosol and was unlikely to have a function in

pur-ine metabolism In this study, the authors failed to

take into account that the A thaliana gene At5g58220

encoded two distinct proteins: OHCU decarboxylase

and TLP Therefore, conclusions drawn from yeast

two-hybrid studies were based on interactions of the

N-terminal region of OHCU decarboxylase with the

receptor kinase brassinosteroid-insenitive-1, rather than

interactions made by TLP Furthermore, their

conclu-sion that the TLP could not be peroxisomal was

largely based on the observation that the TLP did not

possess a C-terminal peroxisomal targeting sequence

Splice variants have been detected for most

eukary-otic TLP genes and some of these variants result in the

truncation of the TLP at the N-terminus This

trunca-tion has no effect on amino acid residues known to be

involved in the function of the protein, but result in

the deletion of the PTS2 nona-peptide In the case of

Mus musculus, transcript data available at RIKEN

Mouse Encyclopedia (genome.gsc.riken.go.jp) suggest

that over 90% of TLP gene transcripts possess the region encoding the PTS2 and were isolated from hepatocytes A small proportion of TLP transcripts (< 10%) do not encode the PTS2 and appear not to

be under tissue-specific regulation Splice variations resulting in deletion of the PTS2 have also been described for plant TLPs [11]

All TLP sequences identified in the Viridiplantae kingdom are encoded by multiple exons [11] For example, the TLP gene from A thaliana is encoded by four exons, the last of which encodes the TLP As pre-viously mentioned, exons 1–3 (motif E) encode a pro-tein from COG 3195, which was recently identified as the enzyme OHCU decarboxylase [12,15] The functional relationship between TLP and OHCU decarboxylase will be discussed below

Evidence for gene duplication

The most primitive organisms found to have a trans-thyretin sequence are the lampreys Petromyzon marinus and Lampetra appendix [16] By contrast, TLP genes have been identified in all kingdoms Given their high degree of sequence similarity, it has been hypothesized that the transthyretin gene arose as a result of a dupli-cation of the TLP gene at some stage in early verte-brate evolution [11] Initial phylogenetic analyses of TLP and transthyretin sequences showed a branching

of transthyretin slightly before the separation of the chordates [17] Subsequent analyses using the recently determined transthyretin sequences from lamprey and recent additions to echinoderm expressed sequence tag (EST) databases, suggest that the TLP gene duplica-tion probably occurred just after the separaduplica-tion of echinoderms (S C Hennebry, unpublished results)

Table 1 Bacteria with multiple copies of TLP genes.

Genes encoding cytoplasmic TLP

Genes encoding periplasmic TLP

Pseudomonas fluorescens Pf5 ATCC BAA-477 Proteobacteria, Gammaproteobacteria 1 2

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Following the gene-duplication event, profound

modifications to the duplicated TLP occurred, leading

to the development of a deep channel into which the

THs 3¢,3,5-triiodo-L-thyronine (T3) and

3¢,5¢,3,5-tetra-iodo-L-thyronine (thyroxine, T4) could bind The

nature of this structural modification will be discussed

below

The function of TLP in purine

metabolism

To date, three studies have been performed examining

the role of TLP in vivo In a study of the A thaliana

TLP, no phenotype was observed when an insertional

mutation was introduced into the TLP gene [14]

How-ever, the lack of phenotype observed may be attributed

to the presence of an additional 5-HIU hydrolase in

plants (see later discussion regarding TLP functional

redundancy) In 2003, Eneqvist et al [10] performed

RNA interference (RNAi) studies in C elegans to

determine a loss-of-function phenotype for R09H10.3

and ZK697.8 TLP genes RNAi-treated worms were

scored for embryonic lethality and for postembryonic

phenotypes (sterility, aberrant morphology,

uncoordi-nated movements, egg-laying defects or slow growth)

No obvious phenotype was detected upon examination

of the gross phenotype of the worms using a dissecting

microscope [10] However, more in-depth examination

into a possible phenotype was not performed For

example, the worms were not subjected to any type of

environmental stress In addition, RNAi was

per-formed using dsRNA for a single TLP gene at a time

As such, the RNAi studies in C elegans may have

been more informative had double-knockdown studies

been performed

A role for TLP in purine metabolism was first

pro-posed in 2001 In an effort to develop a greater

under-standing of purine metabolism in the Gram-positive

bacterium, Bacillus subtilis, Schultz et al [18] generated

a series of insertion mutants One of these mutations

was made in the TLP gene (pucM), which is located

immediately downstream of the gene encoding uricase

The bacteria harbouring this mutation were

character-ized as having a reduced rate of proliferation

(com-pared with wild-type bacteria) on media containing

uric acid as the principal source of nitrogen [18]

Purines are major components of nucleic acids and

nucleotides Subsequently, de novo and salvage

path-ways for purine biosynthesis are important

compo-nents in the metabolism of all organisms The ability

to degrade purine compounds, either aerobically or

anaerobically, has been identified in all kingdoms [19]

The aerobic degradation of purines is dependent on

the oxidation of hypoxanthine and xanthine to uric acid via xanthine dehydrogenase⁄ oxidase (E.C 1.1.1.204 ⁄ E.C 1.1.3.22) In humans, anthropoid apes, birds, uricotelic reptiles and most insects, uric acid is the end product of purine metabolism and is thus excreted [20,21] Most mammals and gastropods fur-ther degrade uric acid to allantoin [20,22], fish and amphibians completely degrade purines to urea, ammonia and carbon dioxide [20,23,24], whilst most plants degrade purines to carbon dioxide and ammonia [25]

Purine oxidation, in particular that of uric acid, is the major route of ureide biogenesis in nature Conse-quently, the enzymes involved in the various stages of purine metabolism have been the focus of much inves-tigation Recently, however, the degradation of uric acid to allantoin has been shown to be more complex than originally thought Previously, it had been assumed that uricase (EC 1.7.3.3) was the sole enzyme responsible for the oxidation of uric acid to allantoin However, Tipton’s group [26] showed that the oxida-tion of uric acid by uricase in fact yields the metastable compound, 5-HIU They observed the spontaneous decomposition of 5-HIU to OHCU within 20 min at neutral pH, followed by the spontaneous decarboxyl-ation of OHCU to racemic allantoin The spontaneous decomposition of 5-HIU results in the generation of numerous free-radical species, which ultimately con-tribute to lipid oxidation [27] Given this fact and the observation that only (S)-allantoin is found in nature, Kahn and Tipton [26] proposed the existence of addi-tional enzymes in the uric acid degradation pathway – first to hydrolyse 5-HIU and second to decarboxylate OHCU to (S)-allantoin

As previously discussed, bacterial TLP genes are fre-quently found in close proximity to the uricase gene and

to another gene encoding proteins belonging to COG

3195 In 2005, Lee et al [28] revealed the ability of recombinant TLP from B subtilis and E coli to specifi-cally hydrolyse 5-HIU Importantly, they demonstrated the inability of human transthyretin to hydrolyse the same compound Ramazzina et al [12] subsequently showed that mouse TLP hydrolysed 5-HIU and that the COG 3195 proteins were responsible for the decarboxyl-ation of OHCU to (S)-allantoin Thus, the pathway of the conversion of uric acid to (S)-allantoin via the three enzymes uricase, TLP (5-HIUase) and OHCU decar-boxylase was revealed (see Fig 3) Whether the three proteins are able to form a multi-enzyme complex remains to be determined One could speculate that the ability to do so would be favourable given the rapid kinetics of spontaneous decomposition of both 5-HIU and OHCU

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To date, the TLP from three bacteria [28–30], one

plant (A thaliana; S C Hennebry, unpublished

results) and two vertebrate species [12,17], have been

analysed for 5-HIU hydrolytic activity and have all

been shown to be 5-HIU hydrolases Thus, a role for

TLP in this purine degradation pathway is evident

throughout evolution In addition, the expression of

the TLP gene in some organisms may be uric

acid-dependent For example, in the Gram-positive

bacte-rium Deinococcus radiodurans, both the uricase and

TLP genes are regulated by a novel uric

acid-respon-sive transcriptional regulator of the MarR family [31]

Given the similarities in the structures of purine

metabolism operons among Gram-positive bacteria, it

is likely that both uricase and TLP genes are similarly

regulated in other bacteria

Interestingly, periplasmic TLPs (those from the

Enterobacteria) have also been demonstrated to have

5-HIU hydrolase activity [28,30] Given that in bacteria,

purine metabolism is localized in the cytosol, it is

pos-sible that the TLP from these organisms acts

indepen-dently of the classical purine catabolism pathway In

addition, no enterobacteria have been found to possess

homologs of OHCU decarboxylase or uricase genes

Therefore, the question arises as to the in vivo role of

periplasmic TLP and whether it is capable of

hydroly-sing compounds other than 5-HIU

The fact that TLP has been demonstrated to

hydro-lyse 5-HIU results in its inclusion in the superfamily of

cyclic amidohydrolases (E.C 3.5.2) Other cyclic

amidohydrolases include hydantoinase, allantoinase

and dihydrooratase [32] Cyclic amidohydrolases share

a number of physicochemical characteristics These

characteristics include quaternary, tertiary, secondary

and primary structure as well as the reliance on a

diva-lent metal cofactor via a conserved metal-binding

motif [33] Studies have also shown the inhibitory

action of some divalent cations on cyclic

amidohydro-lase activity as well as the ability of many enzymes

within this group to bind a variety of cyclic amides

with varying affinities [32] TLP does not appear to

share the classic sequence characteristics of cyclic

amidohydrolases (S C Hennebry, unpublished results) Whilst the E coli TLP was crystallized in the presence of Zn2+, it has been shown that TLP is not a zinc-dependent hydrolase [17]

Structural comparison of Transthyretin and TLP

The 3D structures of transthyretin from various organ-isms have been well characterized The first transthyre-tin crystal structure to be solved (that of human) was published in 1978 [34] The Protein Database (http:// www.pdb.org) contains multiple crystal structure coor-dinates for human transthyretin (including multiple amyloidogenic forms and with various ligands bound) The crystal structures of transthyretin from rat [35], chicken [36] and sea bream [37,38] have also been solved All of these structures demonstrate the remark-able conservation of the prealbumin-like fold (as described by SCOP, http://scop.mrc-lmb.cam.ac.uk), which consists of an eight-stranded b-sandwich (strands A-H) with each sheet adopting a greek-key topology A two-turn a-helix usually (with the exception of chicken transthyretin) exists between strands E and F in trans-thyretin The two transthyretin dimers associate, via nonpolar interactions, between the loops joining stands

G and H with the loops joining strands A and B, mak-ing the transthyretin tetramer a ‘dimer of dimers.’ Recently, the first crystal structures of TLP from various organisms were solved Within a short period

of 3 months, the crystal structures for the TLP from

S dublin (pdb: 2GPZ; [30]), E coli (pdb: 2G2N; [39]),

B subtilis (pdb: 2H0E; [29]) and Danio rerio (zebrafish; pdb: 2H6U; [17]) were solved Remarkably, the struc-tures of these proteins, all tetrameric, showed signifi-cant similarity to the published structures of transthyretin By way of example, a comparison of the structure of S dublin TLP with the structures of trans-thyretin from various organisms is shown in Figure 4 Generally, the structural deviation between TLPs and transthyretins from various organisms is of the same order of magnitude to that within the set of

transthyre-Fig 3 Schematic of the oxidation of uric acid Uric acid is oxidized by uricase to 5-HIU, which is subsequently hydrolysed by TLP (5-HIU hydrolase) to OHCU The enzyme OHCU decarboxylase generates (S)-allantoin (Adapted from [26]).

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tin For instance, the rmsd between equivalent Ca

atoms in the structures of TLPs and human

transthy-retin are 1.0 A˚ and 1.2 A˚ for the monomer and dimer

respectively [17] The rmsd between equivalent Ca

atoms in the structures of transthyretin from various

vertebrates is between 0.34 A˚ and 1.59 A˚ [30]

The main differences between the structures of TLP

and transthyretins are found in the loop connecting

b-strands B and C, which is highly exposed to the

solvent in TLP [17] Interruptions in the b-strands A, G

and H are also observed in TLP structures as a result

of alterations to the formation of hydrogen bonds

between strands The carbonyls of residues V104 and

P105 (zebrafish TLP numbering), in the middle of

b-strand G, do not form hydrogen bonds with the

nitro-gen atoms of H12 and Y116 of b-strand H in TLP

The P105 residue, mainly responsible for the b-strand

irregularities, is invariant in TLP sequences, suggesting

a crucial role for the particular conformation observed

in b-strands A, G and H [17]

Structural nature of the TLP and

transthyretin active sites

One of the striking features of transthyretin is the

cen-tral channel of the protein into which the THs bind

This central channel traverses the entire tetramer It

has previously been postulated [40] and demonstrated

[7,41] that the characteristics of the N-termini of

trans-thyretin from different organisms account for

differ-ences in the affinity of the two main THs (T3 and T4)

to the channel by hindering or allowing greater

accessi-bility

The central channel is also present in TLP, albeit

with quite different structural properties Previously, it

was demonstrated that the regions of greatest

similar-ity between TLP and transthyretin were those forming this central channel, namely motifs A’ and B’ (see Fig 1C) Interestingly, differences between TLP and transthyretin within these motifs also account for sig-nificant physicochemical alterations to the central channel of the protein and provide a structural basis for the differing function compared with transthyretin The presence of a conserved, bulky tyrosine residue at the C-termini of TLP (part of the Tyr-Arg-Gly-Ser tet-rapeptide) causes the central channel to become blocked (see Fig 5A) As a result, the dimer–dimer interface of TLP is characterized by two ‘grooves’ on either side of the protein rather than a central channel [30]

Other key residues at the dimer–dimer interface of TLP include H14, R49 and H106 (B subtilis TLP numbering) [29] An examination of the active site of

B subtilis TLP with the uric acid analogue 8-azaxan-thine bound, reveals that these residues form impor-tant interactions with the ligand (see Fig 5B) Indeed, site-directed mutagenesis studies targeting these resi-dues show that substitution at these sites has profound consequences for the 5-HIU hydrolase activity of the TLP [17,29,30] (and see Table 2)

Mutagenesis of H14 and R49 showed that these resi-dues are the most sensitive to mutation, with H14A, H14N and R49E substitutions abolishing enzyme activity (B subtilis numbering) [29,30] However, the conservative substitution at residue 49 from arginine

to lysine had no effect on activity This suggests the need for a positively charged residue at this site Sub-stitution of H105 and Y118 also significantly reduced enzyme activity, by approximately 90% [30] Deletion

of the C-terminus tetrapeptide Tyr-Arg-Gly-Ser signifi-cantly affected enzyme activity, but it has been sug-gested that S121 does not influence the reaction [29]

Fig 4 Comparison of the tertiary structure

of TLP with transthyretin Stereo diagram showing a superimposition of tetramers of Salmonella dublin TLP (magenta) with trans-thyretin from human (1F41, cyan), rat (1KGI, yellow), chicken (1TFP, orange) and sea bream (1SNO, green) Tetramers were superimposed using the A chain only (Adapted from [30]).

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Interestingly, those residues playing a role in enzyme

activity are 100% conserved in all TLP sequences and

100% substituted in transthyretin (see Table 2)

How-ever, substitutions at position 121 (to threonine or glu-tamate) have been observed None of the mutations affected the tetrameric assembly of the TLP molecule [30] Furthermore, the surface charge of the TLP active site is considerably different from the equivalent region

in transthyretin [29,30] An electrostatically positive groove in TLP contrasts the negatively charged TH-binding site in transthyretin (see Fig 5C)

In summary, a comparison of the catalytic cavity of TLP with the equivalent region of transthyretin (the TH-binding channel) revealed that the TLP cavity is significantly shallower and ‘groove-like’ compared with the deep, hollow channel of transthyretin [30] In par-ticular, the substitution of the C-terminal tyrosine (118) with the much less bulky threonine residue fol-lowing duplication, had a profound effect on the shape

of the channel Loss of the tyrosine residue opened up

A

B

C

Fig 5 The active site of TLP (A)

Compari-son of the ligand-binding cleft at the dimer–

dimer interface in (i) human transthyretin

with (ii) Salmonella dublin TLP Residues

that contribute to the active site are shown.

Hydrogen bonds are shown as broken cyan

lines Thyroxine is shown in stick

represen-tation in yellow For clarity, some elements

of secondary structure are not shown

Resi-dues His6, His95 and Y108 (S dublin TLP

numbering) are equivalent, upon structural

alignment, to Lys15, Thr106 and Thr119 of

human TTR (Adapted from [30].) (B) The

active site of B subtilis TLP with (i) the uric

acid analog, 8-azaxanthine bound and (ii)

showing interacting residues (from [29]).

Note that the active site of the B subtilis

TLP is depicted at 90 to those depicted for

transthyretin and TLP in part A (C) (i)

Elec-trostatic surface potential of human

trans-thyretin with thyroxine bound inside the

negatively charged and deep channel at the

dimer–dimer interface of the protein (ii) The

equivalent region in TLP is shallow and

positively charged (Adapted from [30]).

Table 2 Site-directed mutagenesis of conserved residues in TLP.

Transthyretin

residue

Equivalent

residue in TLP

(S dublin TLP

numbering)

Effect of mutation

on TLP activity Publication

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the central channel of the transthyretin molecule,

allowing for the binding of bulkier ligands such as

THs Superimposition of the dimer–dimer interface of

TLP with that of transthyretin illustrates the

evolu-tionary changes that resulted in the functional

transi-tion of the enzyme into a transport protein

Comparison of structures of TLPs from

various organisms

A comparison of the TLP from three species of

bacte-ria with a vertebrate TLP (zebrafish) shows little

struc-tural divergence Major differences between the

S dublinTLP and zebrafish TLP are found in the

flex-ible portions of strands B and C that protrude towards

the solvent and in the conformation of the long loop

connecting strands D and E [17] Greater differences

are observed between the structures of B subtilis and

zebrafish TLPs: loop B-C is significantly shorter in

B subtilis TLP whilst the loop connecting the short

a-helix to strand F is extended

The active sites of TLPs from prokaryotes and

eukaryotes are nearly identical The location and

ori-entation of the residues present in the catalytic pockets

are well maintained, including the putative main

cata-lytic residues H12 and R52 (zebrafish TLP numbering)

The only significant difference is found in the

C-termi-nal serine residue, which assumed different orientations

in the three structures However, the role of this

resi-due in catalysis has been shown to be negligible [29]

Evolution of TLP function in the

context of urate metabolism

Ramazzina et al [12] eloquently demonstrated the

co-evolution of the three proteins [uricase, TLP

(5-HIUase) and OHCU decarboxylase] involved in the

oxidation of uric acid to allantoin Certainly, the

co-localization of these proteins in the peroxisomes of

metazoan and plant species, and the co-regulation of

TLP genes in some bacteria, suggests a concerted

effort in the rapid generation of allantoin The

co-dis-tribution of uricase, TLP and OHCU decarboxylase

genes in nature reveals that whenever an organism is

found to have a uricase gene, it always has both TLP

and OHCU decarboxylase genes [12] In vertebrates,

the loss of these three genes through evolution is

mir-rored For instance, hominoids lost their ability to

degrade uric acid as the result of the inactivation of

the uricase gene in a primate ancestor, some 15 Ma

[42] In humans, the TLP gene has several inactivating

mutations and the OHCU decarboxylase gene does

not appear to be expressed [12]

Uric acid is a potent antioxidant in biological sys-tems Despite uric acid being the end point of purine metabolism in humans and birds, high levels of allan-toin have been detected in their plasma [43,44] Uric acid chelates transition metal ions (minimizing metal-catalysed oxidation), scavenges hypochlorous acid, is a potent quencher of peroxynitrite and reduces haemo-globin oxidation by nitrite (for a review, see [45]) It has been suggested that in humans and birds, the allantoin generated in these organisms could be a mea-sure of the levels of oxidative stress [44]

The nonenzymatic oxidation of uric acid generates 5-HIU, just as in the uricase reaction As previously discussed, 5-HIU is a highly reactive compound, which, if left to spontaneously decompose, is capable

of forming numerous free-radical species, which ulti-mately contribute to lipid peroxidation [27] Therefore, the rapid elimination of 5-HIU would be advantageous

to the organism Whilst birds have lost functional uri-case and OHCU decarboxylase gene products, TLP transcripts have been detected It is tempting to specu-late that the role of TLP in birds might be to rapidly

‘mop-up’ 5-HIU generated through the nonenzymatic oxidation of uric acid, thereby reducing the potential free-radicals generated when 5-HIU is left to spontane-ously decompose The role of TLP in scavenging 5-HIU clearly warrants further investigation

Purine metabolism occurs in the cytosol of bacteria (for a review, see [46]) The fact that most bacteria possess a cytosolic TLP is consistent with this How-ever, it is not clear what the functional role of a TLP localized to the periplasm might be It is possible that the source of 5-HIU to the periplasm could be from the external environment Interestingly, all bacteria which possess a periplasmic TLP are found to colonize various animals Uric acid is secreted on the surface of mucosal epithelial tissues of all animals as part of the innate immune system [47] and is also thought to act

as a microbicidal agent in these instances Because uric acid can easily permeate the outer membrane of these bacteria, it might be that the TLP located in the peri-plasm acts as a primary defence for the bacterium against oxidized uric acid Alternatively, it could be that 5-HIU is generated in small quantities by the non-specific oxidation of the uric acid by other periplasmic enzymes, such as cytochrome c or peroxidase [48,49]

TLP: an enzyme with functional redundancy?

TLP was not the first protein to be identified as having 5-HIU hydrolytic activity Having hypothesized the need for additional enzymes to contribute to the

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