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Tiêu đề Identification of a novel matrix protein contained in a protein aggregate associated with collagen in fish otoliths
Tác giả Hidekazu Tohse, Yasuaki Takagi, Hiromichi Nagasawa
Trường học University of Tokyo; Hokkaido University
Chuyên ngành Applied biological chemistry; marine biosciences
Thể loại Journal article
Năm xuất bản 2008
Thành phố Tokyo; Hakodate
Định dạng
Số trang 12
Dung lượng 655,36 KB

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By screening a cDNA library of the trout inner ear using an antiserum raised against whole otolith matrix, a novel protein, named otolith matrix macromolecule-64 OMM-64, was identified..

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protein aggregate associated with collagen in fish otoliths Hidekazu Tohse1,2, Yasuaki Takagi2and Hiromichi Nagasawa1

1 Department of Applied Biological Chemistry, Graduate School of Agricultural and Life Sciences, University of Tokyo, Japan

2 Division of Marine Biosciences, Graduate School of Fisheries Science, Hokkaido University, Japan

Organisms can design and shape minerals to the desired

conformation and orientation Such mineral structures

are called biominerals and cannot be formed by any

non-biological environments Calcium carbonate is one

of the most common biominerals, formed mainly by

invertebrates, and has three crystal phases: calcite,

ara-gonite and vaterite Although calcite is the most stable

crystal thermodynamically, many organisms can form

metastable aragonite crystals with desired morphologies

under normal environments of pressure and

tempera-ture It is thought that the morphology and

polymor-phism of biominerals can be controlled by the proteins, polysaccharides and complexes (organic matrices) within the biominerals themselves [1,2]

In the past decade, many proteins have been isolated from various calcium carbonate biominerals, and their roles in the formation of crystal morphologies have been discussed These isolated single proteins have some activity in changing crystal morphologies; how-ever, analyses of the single proteins has not led

to insights into how these morphologies and polymorphisms are formed in the biominerals, as the

Keywords

biomineralization; calcium binding; calcium

carbonate; collagen; otolith matrix

Correspondence

Y Takagi, Division of Marine Bioscience,

Graduate School of Fisheries Science,

Hokkaido University, 3-1-1 Minato,

Hakodate, Hokkaido 041-8611, Japan

Tel ⁄ Fax: +81 138 40 5550

E-mail: takagi@fish.hokudai.ac.jp

Database

Nucleotide sequence data are available in

the DDBJ ⁄ EMBL ⁄ GenBank databases under

the accession number AB213022

(Received 31 December 2007, revised 10

March 2008, accepted 13 March 2008)

doi:10.1111/j.1742-4658.2008.06400.x

In the biomineralization processes, proteins are thought to control the polymorphism and morphology of the crystals by forming complexes of structural and mineral-associated proteins To identify such proteins, we have searched for proteins that may form high-molecular-weight (HMW) aggregates in the matrix of fish otoliths that have aragonite and vaterite as their crystal polymorphs By screening a cDNA library of the trout inner ear using an antiserum raised against whole otolith matrix, a novel protein, named otolith matrix macromolecule-64 (OMM-64), was identified The protein was found to have a molecular mass of 64 kDa, and to contain two tandem repeats and a Glu-rich region The structure of the protein and that of its DNA are similar to those of starmaker, a protein involved

in the polymorphism control in the zebrafish otoliths [So¨llner C, Burgham-mer M, Busch-Nentwich E, Berger J, Schwarz H, Riekel C & Nicolson T (2003) Science 302, 282–286] 45Ca overlay analysis revealed that the Glu-rich region has calcium-binding activity Combined analysis by western blotting and deglycosylation suggested that OMM-64 is present in an HMW aggregate with heparan sulfate chains Histological observations revealed that OMM-64 is expressed specifically in otolith matrix-producing cells and deposited onto the otolith Moreover, the HMW aggregate binds

to the inner ear-specific short-chain collagen otolin-1, and the resulting complex forms ring-like structures in the otolith matrix Overall, OMM-64,

by forming a calcium-binding aggregate that binds to otolin-1 and forming matrix protein architectures, may be involved in the control of crystal morphology during otolith biomineralization

Abbreviations

GST, glutathione S-transferase; HMW, high molecular weight; IPTG, isopropyl-b- D -thiogalactopyranoside; OMM-64, otolith matrix

macromolecule-64; PVDF, polyvinylidene difluoride; TFMS, trifluoromethanesulfonic acid.

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organic matrices are thought to be formed from a

complex of individual matrix proteins For example, in

biomineralization of mollusk shells, which have chitin

as the structural molecule in their EDTA-insoluble

fraction [3], isolated proteins from the EDTA-insoluble

fraction exhibit different actions on the crystal

forma-tion when they are applied to crystal inducforma-tion systems

with framework organic substrates [4–7], indicating

that such proteins may interact with the framework

molecules [8] In biomineral matrices, it is believed that

framework molecules construct the basic scaffold and

form water- or EDTA-insoluble matrices with other

proteins that bind to the frameworks In addition, it is

thought that water-soluble proteins and

polysaccha-rides are bound to the frameworks, possess mineral

(calcium)-binding activity and enable water-gaining

dilatation to form gel-like structure of organic

matrices However, the structural and biochemical

bases of the biomineralization framework and

mineral-associated protein have not been elucidated

On the other hand, vertebrates possess collagens as

a main component of the structural framework In fish

otoliths of the inner ear (a calcium carbonate

biomin-eral of vertebrates), collagen functions as a structural

framework substance [9] The structure of fish otoliths

comprises a tree-ring-like layered biomatrix [10], and

collagen forms the ring structures by periodic

deposi-tion onto the otolith matrix [11,12] A gel-like

struc-ture containing the framework is observed upon

decalcification, suggesting that the otolith matrix is

constructed from large aggregates of framework

mole-cules and mineral-associated molemole-cules

In the majority of biomineral matrices, not just

mollusk shells and otoliths, high-molecular-weight

(HMW, > 100 kDa) proteins are observed by gel

elec-trophoresis These substances may be aggregates of

proteins and polysaccharides, and may play important

roles in formation of the phases and⁄ or morphologies of

the crystals because they consist of acidic glycoproteins

and may construct water-insoluble, gel-like structures in

the biomineral matrices Identifying the proteins that

construct the aggregates is extremely difficult, however,

because these proteins are not separable by gel

electro-phoresis or liquid chromatography In the present study,

we have examined and characterized the proteins that

form these aggregates in fish otoliths We had previously

raised an antiserum against whole otolith matrix

con-taining mainly HMW (> 100 kDa) proteins [13], and

here we used this antiserum to screen an inner ear

cDNA library and thereby clone a cDNA encoding a

protein, named otolith matrix macromolecule-64

(OMM-64), that is contained in a HMW aggregate

in the otolith matrix During characterization of this

protein, we revealed that the aggregate also contains the inner ear-specific collagen otolin-1 [9]

Results

Cloning of cDNA and DNA encoding OMM-64

To obtain cDNA clones encoding proteins contained in the HMW aggregate, immunoscreening was performed using an antiserum that reacts mainly with the aggregate

in the otolith matrix [13] After screening, clones conta-ining omm-64 cDNA were obtained, but the sequence of the 5¢ end could not be determined Therefore, 5¢ RACE was performed In addition, genomic DNA encoding OMM-64 was also obtained by genome walking

Structures of OMM-64 protein and DNA The cDNA cloned had a length of 2776 bp and encodes a protein of 628 amino acids (Fig 1 and sup-plementary Fig S1) The open reading frame is followed by a 3¢ UTR containing a putative polyaden-ylation signal, AATAAA (nt 2747–2752) The relative molecular weights of the precursor including the signal peptide and of OMM-64 without the signal peptide were calculated to be 66 580 and 64 486, respectively, based on the deduced amino acid sequence Sequence analysis showed that OMM-64 has three distinct domains: two tandem-repeat domains of SP(G⁄ E ⁄

R)-Fig 1 (A) Schematic of omm-64 DNA and protein structure Detailed sequences of the mRNA and amino acids are shown in supplementary Fig S1 (the GenBank accession number for

omm-64 mRNA is AB213022) The DNA encoding OMM-omm-64 is split into

23 exons (closed boxes), and several transcription factor-binding sites (closed circles) are predicted to occur in the region 5¢ to the gene OMM-64 has two tandem repeats (R1 and R2) and a Glu-rich region (E-rich) SP, signal peptide (B) Expression of omm-64 mRNA examined by RT-PCR Expression of b-actin mRNA was also exam-ined as an endogenous control S.C., semicircular canal; W muscle, white muscle; R muscle, red muscle; B kidney, body kidney; H kidney, head kidney.

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SDS(T⁄ A)(E ⁄ D) (·6) and MDK(D ⁄ E)D (·5) and a

glutamate-rich region Overall, including these

domains, OMM-64 is rich in acidic residues (Asp +

Glu, 35%) In silico analysis using netphos (http://

www.cbs.dtu.dk/services/NetPhos/) predicted that most

serine residues in tandem repeat 1 are phosphorylated

In the whole sequence, 14% of the amino acids are

predicted to be phosphorylated (66 serines, 19

threo-nines and one tyrosine)

A blastp search using the amino acid sequence of

OMM-64 identified starmaker, a zebrafish otolith

matrix protein that contributes to the regulation of

oto-lith crystal polymorphism [14] Although the identity

between these proteins was only 25%, some distinctive

domains of starmaker are conserved in OMM-64

(sup-plementary Fig S2): an N-terminal sequence containing

signal peptides (Met1–Ala36) is highly conserved, and

two (V⁄ G)TTD sequences found in the tandem repeats

of starmaker are also found in OMM-64 By contrast, a

distinctive sequence that is rich in serine and aspartic

acid in starmaker is not conserved in OMM-64, which

has a glutamic acid-rich sequence instead

A partial sequence of omm-64 mRNA was found in

the GenBank EST database (accession number

CX067293) This rainbow trout mRNA had been

iden-tified by random sequencing analysis of a cDNA

library constructed by suppressive subtraction of

whole-embryo mRNA at late neurogenesis stages

(hindbrain swelling + heart tube with peristalsis) from

that at early neurogenesis stages (neural groove +

50% epiboly), suggesting that omm-64 is expressed in

the early neurogenesis stage of the embryo and is

involved in inner ear development

In omm-64 gene, the sequence encoding OMM-64 is

divided into 23 exons, including two large exons in the

middle region of the ORF and the 3¢ UTR (Fig 1)

This exon⁄ intron structure is highly similar to that of

the starmaker gene (supplementary Fig S2): many

small introns are present in the region encoding the

N-terminal portion of the protein (including the signal

peptide and small tandem repeats), a large exon

com-prises the middle region of the ORF, which encodes

the Glu-rich region in OMM-64 and the Ser⁄

Asp-rich region in starmaker, and the 3¢ UTR is

tran-scribed from a single large exon In addition, all of the

distinctive domains of the proteins are translated from

single exons (supplementary Fig 1A)

Inner ear-specific expression of omm-64 mRNA

Expression of omm-64 mRNA was specific to inner ear

tissues (sacculus and semicircular canals), with the

exception of the ovary (Fig 1B and supplementary

Fig S3) In the inner ear sacculus, strong hybridization signals were detected, mainly in the cells at the periph-ery of the macula and in transitional epithelial cells except mitochondria-rich cells (ion-transporting iono-cytes, Fig 2A,B), which can be distinguished from

Fig 2 Localization of omm-64 mRNA expression in the inner ear sacculus by in situ hybridization (A) Sagittal section of whole sac-culus The regions magnified in (B)–(D) are indicated by boxes (B) Macula (M) and transitional epithelial (TE) regions Intense hybridiza-tion signals were observed in the cells at the periphery of the mac-ula (arrowhead) and in transitional epithelial cells omm-64 mRNA was not expressed in the mitochondria-rich cells (MRC) (C) No hybridization signal was observed in the ventral region of the saccu-lus (D) Hybridization signals were barely detected in the distal region of the sacculus (E) Hematoxylin ⁄ eosin staining of the region shown in (B), to differentiate between transitional epithelial cells (TC) and mitochondria-rich cells (ion-transporting ionocytes), which stain positive with eosin Sense-strand probes did not hybridize

to any regions of the sacculus (data not shown) CT, connective tissue; EL, endolymph region; SqE, squamous epithelial cells.

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other types of cells owing to their large size and shape

and positive eosin staining (Fig 2E), and which, like

chloride cells, have Na+⁄ K+-ATPase activity [15]

Weak expression of omm-64 mRNA was detected in the

sensory epithelium (macula) In the ventral, dorsal and

distal areas of the sacculus, by contrast, mRNA

hybrid-ization signals were barely detectable (Fig 2C,D)

Identification of the calcium-binding domain in

OMM-64

To determine the regions that have calcium-binding

activity, six fusions of GST with recombinant proteins

of OMM-64 (rOMM-64) were produced and applied

to a45Ca overlay assay (Fig 3) Of these recombinant

proteins, rOMM-64-I, III, IV and V, which include the Glu-rich domain, were found to have calcium-binding activity rOMM-64-II and -C and GST were stained red using ‘Stains-all’ and were not detected by 45Ca This result suggests that the Glu-rich domain of OMM-64 has affinity for calcium We cannot con-clude, however, that other regions of the protein do not have calcium-binding activity, because we used recombinant proteins that were not phosphorylated

Characterization of native OMM-64

To characterize the native form of OMM-64, western blotting was performed using anti-rOMM-64-C serum

In the sacculus and endolymph, multiple bands were detected but most of these were non-specific, as assessed by comparison with the preimmune serum; however, a 64 kDa band bound specifically to the anti-serum (Fig 4) In the EDTA-soluble otolith matrix, a diffuse immunoreaction band was observed at

> 100 kDa, but weak non-specific binding around

100 kDa was also detected However, a strong specific reaction in the HMW region was observed in both EDTA-soluble and -insoluble matrices, indicating that OMM-64 may be contained in the aggregate of the HMW proteins described above After digestion of the side chains using deglycosylation enzymes, the intensity

of the immunoreactive band in the HMW region was decreased and a new band was detected at 64 kDa, but only after treatment with heparitinase II (Fig 5A) Although the same band was obtained after digestion

Fig 3. 45Ca overlay analysis of fusions of GST and recombinant

OMM-64 variants (rOMM-64-I-V and -C), containing different

domains of the protein, to determine the calcium-binding domain of

the protein (A) Schematic drawing of the recombinant proteins Six

GST-fused recombinant proteins containing the three distinctive

domains of tandem repeat 1 (R1), the Glu-rich domain (E-rich)

and ⁄ or tandem repeat 2 (R2) were synthesized SP, signal peptide

of the OMM-64 precursor (B) ‘Stains-all’ staining of the

recombi-nant OMM-64 variants separated by SDS–PAGE to detect

nega-tively charged proteins as blue bands (left) and 45 Ca overlay

analysis of the proteins (right) I–V, C and G indicate the respective

recombinant proteins G, GST Calmodulin (C), used as a positive

control, was detected at approximately 17 kDa.

Fig 4 Detection of OMM-64 in the inner ear tissues by western blotting using anti-rOMM-64-C serum In the saccular extract (S) and endolymph (E), OMM-64 bands were observed by both ‘Stains-all staining’ and western blotting (arrowheads) All proteins in the EDTA-soluble (O S ) and -insoluble (O I ) otolith matrix were stained blue using ‘Stains-all’ In these matrices, strong immunoreactions were detected in the high-molecular-weight region (arrows).

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of the sugar chains using trifluoromethanesulfonic acid

(TMSF), the HMW aggregate was not completely

digested even after 30 min of treatment (Fig 5B)

After 15 min, many protein bands were detected by

silver staining, suggesting that several proteins in the

otolith matrix are glycosylated and can be separated

by electrophoresis However, most of these protein

bands disappeared after 30 min of treatment,

indicat-ing that these proteins are damaged by long incubation

with TFMS On the other hand, heparitinase II was

able to completely digest the HMW aggregate

(Fig 5A), and OMM-64 was separated from the

aggre-gate in a concentration-dependent manner (Fig 5C)

These results suggest that OMM-64 is contained in the

otolith matrix aggregate consisting of heparan sulfate

glycosaminoglycans, and can be released from the aggregate by deglycosylation

Localization of OMM-64 in the extracellular matrices

To determine the in vivo localization of OMM-64, immunohistochemistry was performed using anti-rOMM-64-C serum Similar to omm-64 mRNA expres-sion, immunoreactivity was detected in cells at the periphery of the macula and in transitional epithelial cells except mitochondria-rich cells (Fig 6A,B) The basement membranes and connective tissues were im-munonegative We found that OMM-64 accumulates

at the apical membrane in macula (Fig 6A) and in the ring-like structures in otoliths (Fig 6C)

Inner ear-specific collagen otolin-1 is contained in the OMM-64-bound HMW aggregate

To purify the mature form of OMM-64, anti-rOMM-64 affinity beads were allowed to react with saccular and otolith matrix extracts After incubation with the oto-lith matrix extract and stringent washing with acidic glycine, the beads were found to bind a HMW protein and two proteins of approximately 95 and 140 kDa (Fig 7) The HMW protein band reacted strongly with anti-rOMM-64 serum, whereas the other two bands of

95 and 140 kDa immunoreacted with anti-recombinant otolin-1 (rOtolin-1) serum, as previously reported [9,11] These results suggest that the HMW aggregate contains OMM-64 and otolin-1 within the otolith

Fig 5 OMM-64 is contained in the HMW aggregate in the otolith matrix and is excised from the aggregate by deglycosylation using TFMS or heparitinase II (A) Western blotting of EDTA-soluble otolith matrix proteins (OSM) after digestion of polysaccharides by glycopeptidase A (0.5 munits, G), chondroitinase ABC (0.5 units, C), heparitinase II (10 munits, H), hyaluronidase SD (25 munits, Y) and endo-a-N-acethylagalactosaminidase (70 munits, E) The HMW aggregate was digested only by heparitinase II (arrowhead), and a

64 kDa protein band appeared instead (arrow) Some non-specific binding was observed when these enzymes alone were subjected

to SDS–PAGE (Enzyme) (B) Time course of the effect of TFMS treatment on the aggregate and free OMM-64 Although the

64 kDa band was observed by western blotting after treatment with TFMS for at least 5 min (arrow) (aOMM-64), ‘Stains-all’ stain-ing showed that the HMW aggregate was not digested completely even after 30 min of treatment (arrowhead) Silver staining indi-cated that the other proteins may be damaged by the 30 min TFMS treatment (C) Heparitinase II digests the HMW aggregate (arrow-head) and separates free OMM-64 (arrow) in a concentration-dependent manner Bovine serum albumin, which was contained in the enzyme solution, was observed at 66 kDa by both silver and

‘Stains-all’ staining.

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matrix We also observed this interaction when anti-rOtolin-1 affinity beads were used for the same experi-ments However, because non-specific immunoreactive bands were observed in western blotting using anti-rOtolin-1 serum, the 95 and 140 kDa bands could not

be confirmed to be otolin-1 Therefore, MALDI-TOF-TOF tandem mass spectrometry was performed to identify the proteins The tryptic peptide mass finger-printing spectra of these proteins were highly similar (supplementary Fig S4), and both proteins were iden-tified as otolin- by both peptide mass fingerprinting and MS⁄ MS ion searches on the mascot server [16] with high scores Although some differences between the spectra were found, structural differences in these proteins could not be identified

By contrast, the HMW aggregate and otolin-1 bands were separately detected when the beads were reacted with the saccular extract (Fig 7), suggesting that these factors exist independently in the cells and are not

Fig 6 Localization of OMM-64 in the inner ear sacculus by

immu-nohistochemistry (A) Macular region of the sacculus after removal

of the otolith Strongly immunoreactive cells were observed at the

periphery of the macula (arrows) OMM-64 was also detected in

the apical region of the macula (arrowheads) No immunoreaction

was detected in connective tissue (CT) EL, endolymphatic space.

(B) Transitional epithelium observed by differential interference

contrast microscopy Intense signals were observed in the

transi-tional epithelial cells (arrows) but not in the mitochondria-rich cells

(arrowheads) [15] (C) Localization of OMM-64 in the otolith region

observed by differential interference contrast microscopy

OMM-64 was localized in the ring-like structures in the otolith (D) No

immunoreaction was observed in the negative control section of

the otolith region incubated with preimmune serum altered to

pri-mary antibody CT, connective tissue; EL, endolymph region; M,

macula; O, otolith; TE, transitional epithelium (E) Schematic of the

inner ear sacculus containing the otolith, indicating the sections in

(A)–(D).

Fig 7 Separation of native OMM-64, otolin-1 and their complex by co-immunoprecipitation Anti-rOMM-64 or anti-rOtolin-1 affinity beads were incubated with NaCl ⁄ P i (N), saccular extract (S) or EDTA-soluble otolith matrix (O), and specifically bound proteins were subjected to electrophoresis and staining using ‘Stains-all’ Western blotting using anti-rOMM-64 and anti-rOtolin-1 antisera was also performed When the affinity beads were incubated with saccular extract, OMM-64 (arrows) and otolin-1 (arrowheads) bound separately to the beads By contrast, incubation with otolith extract resulted in binding of a complex of the HMW aggregate containing OMM-64 and otolin-1 to the beads.

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associated directly In addition, using anti-rOMM-64

beads, a 64 kDa protein, which was not detected by

gel staining, was detected by anti-rOMM-64 serum

Overall, these data suggest that both OMM-64 and

otolin-1 are contained in the HMW protein aggregate

in the otolith matrix

Discussion

Since the proposal of aragonite crystal induction by

water-soluble organic matrices from aragonite

biomi-nerals [1,2], numerous studies have investigated

pro-teins, mainly in the nacre of mollusk shells, to reveal

how aragonite polymorphs are formed in biominerals

However, no single protein that induces aragonite

formation has been identified, although some reports

have presented evidence that multiple matrix proteins

induce aragonite crystals [4–7] We therefore

exam-ined proteins contaexam-ined in aggregates within the

otolith matrix and identified a protein contained in

the HMW protein–glycosaminoglycan aggregate that

also contains the otolith structural protein otolin-1

This protein may exist freely in the saccular cells and

be incorporated into the HMW aggregate in the

otolith In our previous study, an antiserum raised

against whole EDTA-soluble otolith matrix, which

was used for immunoscreening in the present study,

did not bind to a 64 kDa band, but did bind to

the HMW aggregate in the otolith matrix [13] This

indicates that OMM-64 is not freely localized, but is

contained in the HMW aggregate in the otolith

matrix However, what kinds of molecules are present

in the aggregate in addition to OMM-64, otolin-1

and heparan sulfate, and how these proteins interact,

remains unknown

The protein identified has three distinctive domains:

namely, two tandem repeat sequences and a Glu-rich

region Because repeat 1 may be highly

phosphory-lated, and the Glu-rich region and repeat 2 contain

many acidic residues, OMM-64 may be very acidic

overall and may function in interactions with calcium

and subsequent mineral crystallization Although the

putative isoelectric point of the OMM-64 was

calcu-lated to be 3.5, the mature form of OMM-64 may be

more acidic because it may be highly phosphorylated

Although we determined that the Glu-rich region of

the protein has calcium-binding activity, we could not

confirm whether repeat 1 also has activity because we

used non-phosphorylated recombinant proteins for

the calcium-binding assay Therefore, the functions of

the two tandem repeat domains remain unknown

at present We found starmaker and human dentin

sialophosphoprotein to be homologous proteins to

OMM-64 by blast search (blastp and tblastn) The relationship between starmaker and dentin sialophos-phoprotein has been discussed in detail in a previous report [14] Although some structural similarities in the protein and gene were found between OMM-64 and starmaker (see Results), they may not be ortho-logs because of their relatively low identity (25%) However, their structural similarities may lead to similar functions In fact, knocking down starmaker expression induces a variation in the polymorphism

of otolith crystals, from aragonite to calcite [14] Therefore, OMM-64 and starmaker are thought to be related proteins in terms of both structure and func-tion Although we carried out various blast searches using amino acid, mRNA and genomic DNA sequences as queries, no orthologous gene in any other species was identified So¨llner et al [14] also reported that they could not find an ortholog of star-maker These observations suggest two possibilities: (i) omm-64 and starmaker are orthologous genes that are highly diverged, so the identity of their sequences

is low and orthologs cannot be found, or (ii) they are different but similar genes that are conserved only in species that can form aragonite otoliths At present, however, we are unable to differentiate between these possibilities

We have shown that OMM-64 is contained in the HMW aggregate, which may comprise proteins and heparan sulfate glycosaminoglycan chains, although the structures of the proteins and the glycosaminogly-can chains were not characterized Although it has been suggested that glycosaminoglycan chains are involved in the nanoscale processes of calcium carbon-ate biomineralization by associating with crystals via their sulfates [17], no proteoglycan has been identified

in fish otoliths In mammalian bone, heparan sulfate proteoglycans are localized mainly on the cell surfaces, basement membranes and bone matrix, and are involved in bone formation through regulation of cell differentiation factors such as bone morphogenetic proteins and fibroblast growth factors [18] Similar

to the extracellular matrices in bone, it is conceivable that structural proteins such as collagens may also construct the extracellular matrices in the inner ear by binding to glycosaminoglycans, which can hold water and form gels

The tissue-specific and proximal side-specific distri-bution of mRNA expression and immunolocalization suggests the potential function of OMM-64 In the inner ear sacculus, the otolith is close to the proximal side of the sacculus, and calcification of the otolith occurs mainly at the proximal surface [19] In addition, proteins that may be involved in otolith calcification

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are concentrated in the proximal endolymph [20].

Therefore, the proximal region of the sacculus

pro-duces the otolith matrix proteins [13] and forms the

environment for otolith mineralization In particular,

the cells located at the periphery of the macula may be

specialized for production of the otolith matrix

pro-teins, because these cells are rich in rough endoplasmic

reticulum [13], and two otolith matrix proteins, otolith

matrix protein-1 and otolin-1, are also localized in

these cells [11] Similar to the other otolith proteins,

OMM-64 may contribute to the heterogeneity of the

endolymph chemistry and otolith biomineralization In

the otolith matrix, OMM-64 was localized in ring-like

structures, indicating that OMM-64 is periodically

incorporated into the otoliths The manner of

incorpo-ration may be regulated by the binding activity of

OMM-64 to otolin-1, because periodic expression of

omm-64 mRNA was not observed (data not shown),

which binds to OMM-64 indirectly, is localized in the

ring-like structures [9,11] and its mRNA expression

does vary periodically [12]

During otolith development in zebrafish, the sagitta

(saccular otolith) and lapillus (utricular otolith), both

of which composed of aragonite, are formed in the

single otosac of the inner ear at early developmental

stages [24–30 hours post fertilization (hpf)] [21] By

contrast, the vaterite asteriscus develops in the lagena,

which differentiates from the otosac after initiation of

the formation of sagitta and lapillus [15 days post

fer-tilization (dpf)] [22] Therefore, it is possible that the

developmental process that underlies aragonite

oto-liths and vaterite otooto-liths is different Otolin-1, which

is necessary for aragonite crystal formation in vitro, is

expressed in the early stages of development of the

inner ear (48 hpf) and is involved in the seeding

and⁄ or nucleation of the sagitta and lapillus [23] On

the other hand, omm-64 may be expressed at earlier

stages because mRNA expression was found in the

trout embryo at the 50% epiboly stage (see Results)

If omm-64 mRNA is really expressed at this stage, it

represents the earliest known expression of all inner

ear-specific marker genes found to date Otolith nuclei

are formed at about 24 hpf by aggregating proteins

and polysaccharides secreted from epithelial cells [24]

Starmaker is also expressed at an early stage (24 hpf)

[25] Therefore, OMM-64 and starmaker are likely to

be contained in the aggregate and contribute to

for-mation of the aragonite polymorph

In summary, we have identified a novel protein,

OMM-64, contained in the HMW aggregate in the

otolith matrix, and shown that the aggregate also

con-tains ear-specific collagen, otolin-1, and forms

frame-work mineral constructs The two proteins, OMM-64

and otolin-1, are expressed in the same cells in the inner ear sacculus and are secreted into the extracellu-lar matrices of the inner ear In the otoliths, they are both localized in the ring-like structures These find-ings identify for the first time proteins with these func-tions that construct matrix aggregates in calcium carbonate biominerals

Experimental procedures

Animals

Rainbow trout (Oncorhynchus mykiss) weighing approxi-mately 1000 g were used They were reared in outdoor ponds at 10–15C under natural light for at least 10 days before collection of the samples

Cloning of cDNA and DNA encoding OMM-64

As described previously [13], we detected HMW proteins that may be aggregated in the otolith matrix by western blotting using an antiserum raised against whole water-sol-uble otolith matrix To identify proteins contained in these aggregates, immunoscreening of a cDNA library was per-formed using this antiserum Approximately 200 000 clones contained in a kZAP II (Stratagene, La Jolla, CA, USA) inner ear cDNA library, constructed according to the method described by Murayama et al [26], were grown on

each clone were induced and transferred to poly(vinylidene difluoride) (PVDF) membranes (Millipore, Billerica, MA, USA), which had been soaked with 20 mm

blocking with 5% fat-free dried milk in NaCl⁄ Tris (50 mm

were incubated with the antiserum at a 1 : 1000 dilution overnight Each immunoreacted membrane was then incu-bated with secondary antibody (horseradish peroxidase con-jugated anti-rabbit IgG; Bio-Rad, Hercules, CA, USA) at a

1 : 1000 dilution for 2 h Immunoreaction was detected by catalysis of the substrate diaminobenzidine diluted in

proteins were selected, and the pBluescript phagemids were excised using ExAssist helper phage (Stratagene) according

to the manufacturer’s protocol To determine the 5¢ end of the cDNA, 5¢ RACE was performed using a SMART RACE cDNA amplification kit (Clontech, Mountain View,

CA, USA) with reverse primer 5¢-GTGACAACATTGTGA TGGGATAGTTT-3¢ (nt 79–54)

After cDNA cloning, the sequence of the genomic DNA encoding OMM-64 was determined by PCR-based cloning

To determine the internal introns, PCR was performed using gene-specific primers designed according to the sequence of omm-64 cDNA A GenomeWalker universal kit

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(Clontech) was used to clone the region 5¢ to the gene.

PCR products were ligated into pGEM-T Easy vector

(Pro-mega, Madison, WI, USA), and the ligated plasmid DNAs

were transformed into XL1-blue-competent cells

(Strata-gene) After growth and harvest of the Escherichia coli cells,

the amplified DNAs were recovered using a QIAprep

mini-prep kit (Qiagen, Hilden, Germany) and sequenced using a

DNA sequencer (3130xl Genetic Analyzer; Applied

Biosys-tems, Foster City, CA, USA)

Expression analyses of omm-64 mRNA

Total RNA was isolated from various organs (see Fig 2)

using ISOGEN (Nippon Gene, Tokyo, Japan), and treated

con-tamination in the total RNA was confirmed by lack of

amplification of a b-actin mRNA fragment by PCR using a

pair of primers (5¢-ATCACCATCGGCAACGAGAG-3¢

reverse transcription After purification using phenol⁄

chlo-roform, 1 lg of the total RNA was reverse-transcribed

using a first-strand cDNA synthesis kit (Amersham

Bio-sciences, Little Chalfont, UK) Using 1⁄ 100 aliquots of the

first-strand cDNAs as templates, PCR was performed using

2385–2407) and 5¢-GCGTCATTAAACGTATGTACACT-3¢

(nt 2600–2578) Expression of b-actin mRNA was verified

using the primers described above

For in situ hybridization, a 216 bp fragment (nt 2385–

2600) of omm-64 cDNA was amplified by PCR as described

above and ligated into pGEM-T vector (Promega) The

plasmid DNA was digested with NotI or NcoI, and

anti-sense and anti-sense probes labeled with digoxigenin were

pro-duced by in vitro transcription using T7 and SP6 RNA

polymerase (Roche, Mannheim, Germany) The specificity

of the probes was confirmed by northern hybridization

(supplementary Fig S1), and omm-64 mRNA expression in

the paraffin sections of the sacculi was detected by in situ

hybridization as described previously [27]

For northern blotting analysis, total RNA of the inner

ear sacculus and ovary (10 lg each) extracted using

ISO-GEN (Nippon Gene) and the sense- and antisense-strand

RNA probes (0.1 lg each) produced as described above

were subjected to 1.2% agarose gel electrophoresis After

electrophoresis, the RNAs were blotted onto a

hybridized with digoxigenin-labeled sense- and

After washing the membrane twice each with 2· SSC and

0.1· SSC at 68 C for 30 min each, hybridization signals

were detected by immunodetection using alkaline

phospha-tase-conjugated anti-digoxigenin Fab fragments (Roche)

coupled with CDP-star alkaline phosphatase substrate

(Roche) according to the manufacturer’s protocol

Determination of the calcium-binding domain using recombinant OMM-64 variants

Six recombinant fusion proteins comprising glutathione S-transferase (GST) and various regions of OMM-64

Ala21–Ser628; rOMM-64-V, Arg410–Ser543; rOMM-64-C,

regions of the omm-64 cDNA were amplified by RT-PCR using six pairs of primers (rOMM-64-I, 5¢-CGCGGATCC ACCGTAGACACTTATGATATA-3¢ and 5¢-CGCCTCCA CCTAAGAGGCATCCTTGTCCAC-3¢; rOMM-64-II,

5¢-CGCCTCGAGCTAAGAGTCAGCTTGCACGTC-3¢; rOMM-64-III, 5¢-CGCGGATCCGCTGATGTGACCAGT GATGAC-3¢ and 5¢-CGCCTCGAGCTATTTGGGCTCTT TCATCAT-3¢; rOMM-64-IV, 5¢-CGCGGATCCGCCCCT GTTAATGATGGAACC-3¢ and 5¢-CGCCTCGAGCTAA GAAGACTGGGCTGCCAG-3¢; rOMM-64-V, 5¢-CGCGG ATCCAGGCAAGATTTTAAGCATCCA-3¢ and 5¢-CGCC TCCACCTAAGAGGCATCCTTGTCCAC-3¢;

rOMM-64-C, 5¢-CGCGGATCCGACTCAGTGGATGACCAATCC-3¢

AG-3¢) that had 5¢ adapters corresponding to BamHI (GGATCC) and XhoI (CTCGAG) restriction sites, respec-tively In the reverse primers, stop codons (TAG) before the XhoI sites were also added PCR products were doubly digested by the restriction enzymes, purified using a QIAquick PCR purification kit (Qiagen), and ligated into pGEX 6p-1 vector (Amersham Biosciences), which had been digested and purified in the same way as the PCR products After transformation into XL1-blue cells and confirmation of the sequences, the plasmid DNA was trans-formed again into BL21 E coli cells (Amersham Biosciences)

200 lL of the cells was transferred to 20 mL of new LB

The GST–recombinant protein fusions were then induced

by addition of IPTG at a final concentration of 1 mm After incubation at 37C for 2 h, the cells were collected

by centrifugation at 3000 g for 10 min and lysed in 10 mL

of extraction buffer (5 mm EDTA, 0.5% Triton X-100,

then centrifuged at 30 000 g for 10 min and the supernatant was collected We added a 0.5 mL bed of glutathione– Sepharose beads (Amersham Biosciences) equilibrated with

1.8 mm KH2PO4, pH 7.4) to the extract, and allowed the GST–recombinant protein fusions to bind to the beads at

10 mL of extraction buffer and NaCl⁄ Pi

directly applied to SDS–PAGE under reducing conditions

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Separated proteins were stained with ‘Stains-all’ (Sigma,

St Louis, MO, USA) [28] or were blotted onto a PVDF

membrane to detect45Ca2+-binding activity [29]

Production of antibody against recombinant

OMM-64

The recombinant rOMM-64-C protein that bound to

glutathione–Sepharose beads was digested from GST by

Bio-sciences) at 4C for 2 days, eluted with 1 mL of NaCl ⁄ Pi,

and concentrated and desalted using Ultrafree cartridges

(Millipore, 5000 Da cut-off) The digested rOMM-64-C was

completely separated from GST using a Sep-Pak Cartridge

C18 column (Waters, Millford, MA, USA) by stepwise

elution with acetonitrile

rOMM-64-C had been confirmed by MALDI-TOF mass

spectrometry (4700 Proteomics Analyzer, Applied

Biosys-tems), the buffer of the protein was changed to NaCl⁄ Pi

using Ultrafree cartridges (5000 Da cut-off) Production of

rabbit antiserum raised against the recombinant protein

and affinity purification of the antibody were performed by

Hokkaido System Science (Hokkaido, Japan)

Collection of inner ear proteins

Dissection of whole inner ear and collection of the

endo-lymph and otoliths were performed as previously described

[30] After the endolymph and otoliths had been collected,

the sacculi were washed three times with 0.9% NaCl and

homogenized in the same solution The homogenate and

endolymph were centrifuged at 100 000 g for 1 hr and the

supernatants were collected Otoliths were washed

vigor-ously five times each for 1 min each with 1% SDS and

dis-tilled water, and were immediately decalcified in 0.5 m

was changed every day by centrifugation at 25 000 g for

10 min, and the supernatant was collected The

superna-tants were stored at)30 C After complete decalcification

(approximately 5 days), the stored solutions were

concen-trated and the solvent was changed to 20 mm Tris⁄ HCl

(pH 8.0) using Ultrafree cartridges (5000 Da cut-off) The

EDTA-insoluble matrix (the pellet from the final

centrifu-gation of the EDTA-decalcified solution) was washed five

times with 20 mm Tris⁄ HCl (pH 8.0) and the proteins were

extracted by boiling in denaturing solution (8 m urea,

Tris⁄ HCl pH 8.0) for 10 min

Analyses of protein profiles

SDS–PAGE was performed under reducing conditions

After separation of the proteins, the gels were stained with

‘Stains-all’ [28] or silver to detect negatively charged pro-teins and all propro-teins, respectively

To detect OMM-64 and otolin-1 by western blotting, anti-rOMM-64-C and anti-recombinant otolin-1-C [9] sera were used Ten micrograms of protein extracted from inner ear was separated by SDS–PAGE and blotted onto a PVDF membrane The membrane was incubated first in 5% fat-free dried milk in NaCl⁄ Tris for 1 h, and then in the same solu-tion containing the antibodies (1 : 1000 dilusolu-tion) overnight After washing the membrane twice (10 mins each) with NaCl⁄ Tris containing 0.1% Tween-20 and once with NaCl ⁄ Tris, specific binding of the antibodies was detected by using Supersignal West Femto Maximum Sensitivity Substrate (Pierce, Rockford, IL, USA), and the corresponding second-ary antibody (horseradish peroxidase-conjugated anti-rabbit IgG, 1 : 5000), according to the manufacturer’s protocol

Deglycosylation of proteins

Ten micrograms of otolith matrix protein were desalted in

an Ultrafree cartridge (5000 Da cut-off) and completely dried in a centrifugal concentrator (VC-96W, Taitec, Saitama, Japan) Chemical deglycosylation of the proteins was performed by incubation with 50 lL of trifluorome-thanesulfonic acid (TFMS) at 0C for 0, 5, 15 and 30 min The solutions were neutralized by adding 500 lL of ice-cold buffer (1 m Tris) The sample solvent was changed to

(5000 Da cut-off) For enzymatic digestion, 10 lg of water-soluble otolith matrix protein, completely dried in a

10 lL of the following deglycosylation enzymes dissolved in buffers described in the manufacturer’s protocol (Seika-gaku, Tokyo, Japan): glycopeptidase A (0.5 munits), chon-droitinase ABC (0.5 units), heparitinase II (0, 1, 2, 3, 5 and

10 munits), hyaluronidase SD (25 munits) or endo-a-N-ac-ethylgalactosaminidase (70 munits) The samples were sub-jected to 10% SDS–PAGE, and OMM-64 was detected by western blotting as described above

Immunohistochemistry

Paraffin sections (5 lm) of the sacculi containing otoliths were produced as previously described [13] After de-par-affination and rehydration, the sections were incubated at

(1 : 1000) for 1 h and then with anti-rOMM-64 serum

immunoreaction with secondary antibody (1 : 2000, peroxi-dase-conjugated anti-rabbit IgG, Bio-Rad) for 2 h, the sec-tions were washed three times with NaCl⁄ Pi, and specific binding of the antibody was detected by incubating the sections in a solution of 10 mm diaminobenzidine in

10 mm NaCl⁄ Pi

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