rapid hyaluronan uptake assoclated with

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RAPID HYALURONAN UPTAKE IS ASSOCLATED WITH ENHANCED MOTILITY AND TRANSFORMATION: IMPLICATIONS FOR AN INTRACELLULAR MODE OF ACTION Lisa Janine Collis A thesis submitted in conformity with the requirements for the Degree Master of Science, Graduate Department of Anatorny and Ce11 Biology University of Toronto O Copyright by Lisa Janine Collis, 1999

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Page 1: RAPID HYALURONAN UPTAKE ASSOCLATED WITH

RAPID HYALURONAN UPTAKE IS ASSOCLATED WITH ENHANCED MOTILITY AND TRANSFORMATION:

IMPLICATIONS FOR AN INTRACELLULAR MODE OF ACTION

Lisa Janine Collis

A thesis submitted in conformity with the requirements

for the Degree Master of Science,

Graduate Department of Anatorny and Ce11 Biology

University of Toronto

O Copyright by Lisa Janine Collis, 1999

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National Library 1+1 of Canada Bibliothèque nationale du Canada

Acquisitions and Acquisitions et Bibliographie Services services bibliographiques

395 Wellington Street 395, nie Wellington OttawaON K1AON4 Onawa ON K1AON4 Canada Canada

The author has granted a non- exclusive licence allowing the National Library of Canada to reproduce, loan, distribute or sel1 copies of this thesis in rnicroform, paper or electronic formats.

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The author retains ownership of the L'auteur conserve la propriété du copyright in ths thesis. Neither the droit d'auteur qui protège cette thèse. thesis nor substantial extracts fiom it Ni la thèse ni des extraits substantiels rnay be printed or othewise de celle-ci ne doivent être imprimés reproduced without the author's ou autrement reproduits sans son permission. autorisation.

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Rapid Hyaluronan Uptake is Associated with Enhanced Motility and

Transformation: Implications for an Intracellular Mode of Action

Lisa Janine Collis

For the Degree Master of Science, 1999

Department of Ariatomy and Cd1 Biobgy

University of Toronto

ABSTRACT

Transformed fibroblasts rapidly intemalize Texas Red labeled hyaluronan (TR-

HA), and target it to ce11 nuclei. processes and the pennuclear region. TR-HA uptake is

blocked by: excess polymeric HA, HA-dodecascuicharide, colchicine, BrefeldinA, and by

digestion of TR-HA with hyaluronidase. Uptake is dose dependent, saturable and strongly

enhanced upon ceIl attachment, monolayer wounding and transformation. TR-HA nuclear

targeting requires a minimum of 30 HA-saccharides, suggesting the existence of novel

hyaladherins. Enhanced uptake correlates with increased CD44 and RHAMM expression

and both CD44 antibodies and RHAMM peptides alter uptake. Interestingly, MEK

inhibition also affects uptake. Connexin43 overexpression, regulated by RHAMM

expression, enhances HA uptake and targeting to cell processes. In vitro, HA enhances

DNasel-DNA interaction, suggesting nuclear HA'S potential influence on gene expression.

Roles for intracellular hyaluronan in cell function are suggested by a correlation of ce11

process HA targeting with increased motility, after oncogene transformation, monolayer

u-ounding, or phorbol ester treatment.

Page 4: RAPID HYALURONAN UPTAKE ASSOCLATED WITH

I would like to express the deepest admiration and thanks to my parents who are a conrinual

source of love and support and to Chad for his love, patience and constant encouragement,

and it is to thent that I dedicate this thesis.

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This thesis would not have been completed without help and support from

numerous CO-workers and friends.

1 would first like to thank my supervisor Dr. Eva Turley, for providing me with the

opportunity to do this work, and for her guidance and support throughout this study. 1 am

likewise indebted to our collaborators Dr. Glen Prestwich and Michael Ziebell of the

University of Utah for their assistance in developing and analyzing the TR-HA conjugate,

Dr. Markku Tammi and Dr. Raija Tammi of the University of Kuopio, Finland for

providing me with purified and labelled HA oligosaccharides, Dr. Jim Nagy of the

University of Manitoba for providing the connexin43 transfected glioma cells and Bruce

Lynn of the University of Manitoba for his patience during our attempts at live uptake, and

his instruction on image analysis. 1 further wish to express my sincere gratitude to my

cornmittee members Dr. Michael Opas and Dr. Sean Egan for their time and knowledge.

1 am also indebted to ail the members of Dr. Turley's research group, past and

present. My thanks extend especially to Frouz Paiwand and Rene Harrison who have

been such great benchmates, confidants and friends during this formative experirnce. 1

would also like to express my thanks to Jingbo A, Shiwen Zhang, Arian Khandani, Jie

Lu, FuSheng Wang, Joycelyn Entwistle and Judy Edwards for their support as both

friends and colleagues.

This study was performed at The Hospital for Sick Children in the Department of

Cardiovascular Research under the aegis of the Department of Anatomy and Cell Biology

of the University of Toronto during the yean 1997 - 1999 with financial support from an

MRC studentship and Hyal Pharrnaceutical Corporation.

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TABLE OF CONTENTS

ABSTRACT ...............................................................................................................

DEDICATION ...........................................................................................................

ACKNO WLEDGMENTS .........................................................................................

TABLE OF CONTENTS ..........................................................................................

LIST OF FIGURES ...................................................................................................

LIST OF ABBREVIATIONS ...................................................................................

1 . INTRODUCTION ..............................................................................................

.................................................................. . 1.1 Biochemical Properties of HA

1.1 . 1. Structure .....................................................................................

................................................... 1.1.2. Family of glycosaminoglycans

1.1.3. Synthesis of HA ........................................................................

1.1.4. HA degradation ...........................................................................

1.2. Functions of HA ..................... .... ............................................................

.............................................. 1.2.1. Physicochemical funciions of HA

..................................... 1.2.2. HA binding proteins - the hyaladherins

1.2.2.1. CD44 ...........................................................................

..................................... 1.2.2.2. RHAMM and related proteins

L2.3. Hyaluronan and tumorigenesis ...................................................

.................................................. L2.3. Hyaluronan and wound healing

1.3. Rationale of this study ...............................................................................

II . MATERIALS AND METHODS .......................................................................

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II.2. Conjugation of HA with Texas Red ........................................................

II.3. Treatment of cells with TR-HA rind analysis of resultant intracellular

fluorescence ...........................................................................................

Digestion of TR-HA with hyaluronidase ................................

Cornpetition with unlabeled HA and HA oligosaccharides ....

Treatment of celis with Texas Red probe alone ......................

Treatment of cells with Syto16 ...............................................

.................................................. TR-HA concentration curves

............................ TR-HA treatment of cells over timecourses

Prebinding of peptides to TR-HA ...........................................

................. Anti-CD44 and Anti-RHAMM antibody blocking

.......... AMAC labeled HA-oligosaccharide treatment of cells

II.4. Wounding assays ......................................................................................

................................................................................ fI.5. Immunofluorescence

U.6. Flow cytometry (FACS analysis) .............................................................

...................................................................................... IT.7. DNaseI digestions

III . RESULTS ............................................................................................................

El . 1 . Texas Red labeled hyaiuronan (TR-HA) is rapidly taken up into ras-

transforrned cells ..................................................................................

III.2. TR-HA uptake is dose and time dependent and increases with cellular

transformation ......................................................................................

............................ OI.3. Specific HA oligosaccharides inhibit TR-HA uptake

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m.4. Thiny HA-saccharide minimum length is required for effective

targeting of HA to the nucleus .............................................................

m.5. Anti-CD44 antibodies affect TR-HA uptake .........................................

iiI.6. Subcellular distribution of RHAMM is dramatically altered upon HA

............................................................................................... treatmen t

m.7. RHAMM peptides block TR-HA targeting to the nucleus .....................

iII.8. TR-HA uptake is unaffected by blocking with RHAMMexon9

antibody ................................................................................................

IU.9. HA uptake varies depending upon length of ce11 plating time and

......................... positively correlates with functional RHAMM levels

III . 10 . Connexin43 transfected cells show incmased TR-HA uptake and

.................................................... targeting to the tips of ce11 processes

iII . 1 1 . TR-HA uptake is decreased upon MEK inhibition ...............................

III . 12 . HA is targeted to the ce11 processes upon phorbol ester treatment of

................................................................... parental iOT112 fibroblasts

III . 13 . TR-HA shows enhanced uptake and tatgeting to the tips of the ce11

processes at the wound edge in parental IOT112 fibroblasts ................

IV . DISCUSSION ......................................................................................................

IV . 1 . The phenornenon of rapid HA uptake ....................................................

IV.2. Mechanisms of HA uptake .....................................................................

N.2.1. The potentiai role of ce11 surface receptors in TR-HA uptake ...

N.3. Potential roles for intracellular targeting of HA .....................................

TV.3.1. Potentiai role of HA in the nucleus ..........................................

vii

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......................... N.3.2. Potential roles of HA uptake in transformation

................... N.3.3. Potential roles of intracellular HA in ce11 motility

iV.4. Conclusions ............................................................................................

V . REFERENCES ....................................................................................................

VI . APPENDIX 1 - ADDITIONAL RESULTS SECTION ....................................

VI . 1 . Resul ts ......................................................................................................

........ . VI 1.1. Colchicine treatment prevents nuclear uptake of TR-HA

VI . 1.2. Brefeldin A pretreatrnent blocks uptake of TR-HA by

....................................................... 10T 1 /2 parenta1 fi broblasts

.............. . VI 1.3. HA alter'; the ability of DNaseI to interact with DNA

VI.2. Discussion ................................................................................................

..................................... Vf.2.1. Cytosolic factors in the uptake process

........ VI.2.2. Potential influence of HA on DNA - protein interaction

................... ................... VIL APPENDIX 2 - HYALURONAN DETECTION ..

......... ...................................... . VI 1.1. Existing probes for hyaluronan ..

.................................... . VI 1.2. The Texas Red - hyaluronan conjugate

viii

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LIST OF FIGURES

1 . INTRODUCTION

..................................................................... Figure 1.1. Structure of hyaluronan 4

Figure 1.2. Extrusion mode1 for HA synthesis ................................................... 6

Figure 1.3. Hyaluronan binding proteins - the hyaladherins ............................... 14

II . METHODS

. . Figure II 1 Conjugation of HA to Texas Red hydrazide ..................................... 30

III . RESULTS

Figure UI . 1 . TR-HA is targeted to the nucleus. perinuclear area. and ruffles of

.................................................................. ras transformed ce1 1s

Figure III.2. TR-HA and Syto l6 are found in the nucleus in the same focal

............................................................................................. plane

Figure m.3. TR-HA uptake is HA specific ............................................................

Figure m.4. TR-HA uptake is dose and time dependent. and increased in

................................................................. transformed ceIl lines

................................... Figure m.5. Oligosaccharides of HA block TR-HA uptake

Figure m.6. AMAC labeled Oligosaccharides of HA can be targeted to the

perinuctear and nuclear compartments of cells in a size

dependent manner ......................................................................

Figure iIi.7. CD44 mAbs alter TR-HA uptake .......................................................

Figure III.8. Subcellular distribution of RHAMM is dramatically aitered upon

HA treatment ..............................................................................

Figure III.9. RHAMM peptides block TR-HA targeting to the nucleus .................

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Figure III. 10 HA uptake is unaffected by blocking with RHAMMexon9

an tibody . . . . . . . . .. . . . . .. . .. .. . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . .. . . . . . . . . . . . . .

Figure m. 1 1 HA uptake varies depending upon length of ceil plating time and

positively correlates with functional RHAMM levels ...............

Figure III. 12 Connexin43 transfected cells show increased TR-HA uptake and

targeting to the tips of ceIl processes ..........................................

Figure III. 13 TR-HA uptake is decreased upon MEK inhibition ............................

Figure III. 17 HA is targeted to the ce11 processes upon phorbol ester treatment

of parental 1 OT 1 /2 fibroblasts . . . .. . . . . . . . . . .. .. . . . .. . . . .. . .. . . . . . . . . .. . . . . . . ... . .

Figure m. 18 TR-HA shows enhanced uptake and targeting to the tips of the ce11

processes at the wound edge in parental lOT112 fibroblasts .......

VI. APPENDIX 1

Figure VI. 1 Colchicine treatment prevents nuclear uptake of TR-HA .................

Figure V1.2 Brefeldin A pretreatment blocks uptake of TR-HA by 10T 112

parental fibroblasts ..... . .. .. . . . . . . .. . . . . ... .. . . . .. . . . .. . . . .. . . . . .. . . . .. . .. .. . . . . . . .. .

Figure VL3 HA alters the ability of DNaseI to interact with DNA .......................

Figure V1.4 Mode1 of potential uptake mechanisms and intraceliular functions

of HA .........................................................................................

VII. APPENDIX 2

Figure VU. 1 .HPLC analysis of TR-NA conjugate ................................................

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aa

BSA

CD44

CD44v

cDNA

CPC

CS

Da

DMEM

DMSO

ECM

EDTA

ERK

FACS

FCS

FITC

GAG

GPI

HA

HA#

HABP

HARLEC

HAS

Amino acid(s)

Bovine serum albumen

Standard CD44 isoform

Variant CD44 isoform

Complimentary DNA

Cetylpyridinium chloride

Chondroitin sulphate

Dalton

Dulbecco's modified eagle's medium

Dimethyl sulfoxide

Extracellular matrix

Ethylenediaminetetraücetate

Extracellular signal regulated protein kinase

Fluorescence activated ce11 sorter (used in flow cytometry)

Fetal calf semm

Fluorescein isothiocyanate

Gl ycosaminogl ycan

Glycosylphosphatidyl inositol

Hyaluronan, hyaiuronic acid, hyaluronate

Hyaluronic acid oligosaccharide of "#" monosaccharide units

Hyaluronan binding protein(s)

Hyaluronan receptor for liver endothelial cells

Hyaiuronan synthase

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HBSS

HMW

HS

IgG

kb

kDa

LMW

m Ab

MAP

MAPK

MEK

MW

MWCO

PA"

PBS

PDGF

PKC

PMA

rER

RHAMM

RHAMMA 1 -5

RHAMMexon9

RITC

TR

TR-HA

Hank's batanced salt solution

High molecular weight

heparin sulphate

Immunoglobulin G

Kilobase(s) or lOOObp

Kilodalton or 1 OOOda

Low molecular weight

Monoclonal anti body

Mitogen associated protein

Mitogen associated protein kinase

MAPWERK kinase

Molecular weight

Molecular weight cut-off

Polyclonal antibody

Phosphate-buffered saline

Platelet derived growth factor

Protein kinase C

Phorbol rnyristate acetate

Rough endoplasmic reticulum

Receptor for hyaluronan mediated motility

RHAMM exons 1-5 deleted (old nomenclature WAMMv4)

old nomenclature = exon4

Rhodarnine isothiocyanate

Texas red

Texas red - hyaluronan conjugate

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CHAPTER 1

INTRODUCTION

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Hyaluronan (HA) was first descnbed as a polysaccharide isolated from the

vitreous humour in 1934 (Meyer et al. 1934). It contained uronic acid and Meyer

named the polysaccharide hyaluronic acid from hyulos (glassy, vitreous) and uronic

acid. However at physiological pH al1 carboxyl groups on the uronic acid residues

are dissociated and the polysaccharide has therefore been named sodium hyaluronate

when sodium is the counter ion. It is often difficult to specify the counter ion, for

example in a tissue, and Balzacs et al. (1989) therefore suggested that the name

hyaluronan (HA) should be used.

Hyaluronan is a molecule of paradoxes and contrasts. On the one hand, it is

a homopolymer of simple disaccharide units endlessly repeated. present in some

tissues (such as the cockscomb and vitreous humor) in large amounts and

performing very simple mechanical jobs as a space filler or lubricant (in synovial

joints). On the other hand, HA takes part in ce11 surface phenornena of great

specificity when used at very low dilution in the presence of specific receptors.

Interest in HA has increased dramatically since 1980 when major clinical

applications in optharnology and in the treatment of joint disease were introduced.

Since then, HA has found multiple applications in drug delivery and surgery. HA

has been found to enhance absorption of drugs and proteins through mucus tissues

(Morimoto et al. 199 1). When combined with HA, the efficacy of many agents

(such as anti-inflammatory drugs) are strengthened (Goa and Benfield 1996; Miller

et al. 1997). HA has found important applications in the field of viscosurgery,

viscosupplementation and wound healing (Davidson et al. 1991; Juhlin et al. 1997).

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in reproductive medicine, HA is used to improve retention of the mobility of

cryopreserved and thawed spermatozoa (S bracia et al. 1997).

HA is a ubiquitous compound and there is no known genetic disease in

which HA is not synthesized. HA may therefore be of fundamental importance in

the animal organism and mutations causing defects in HA synthesis may be lethal.

HA is found in virtually every species in the animal kingdom, in every tissue in the

human body as well as the capsule of certain microorganisms. Moreover the HA

repeating disaccharide is identical in al1 tissues and species and consequently HA is

never recognized as immunologicdly foreign (Fraser and Laurent 1997).

1.1. Biochemical Properties of HA

1.1.1. Structure

The polysaccharide HA is a negatively charged linear pol ymer with the

structure 143-4 D-glucuronic acid ( 1 -[P-3) N-acetyl-D-glucosamine ( 1 -1,-P-4)

(Figure 1.1.) that can have a molecular mass of up to several million daitons. The

relative rnolecular weight of HA varies dramatically depending upon its source. For

example, the HA isolated from human synovial fluid is about 7000kDa, from

human blood serum it is approximately 200kDa, and from human urine it rnay be as

small as 4- 12kDa (Fraser and Laurent 1996).

Studies on conformation of HA by X-ray diffraction and spectroscopy

indicate that the molecule c m take up helical conformations stabilized by hydrogen

bonds that run in parallel with the chah axis (Scott and Tigwell 1978; Scott 1989).

The polyrner consequently takes up a stiffened helical configuration which gives the

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molecuie an overail expanded coi1 structure in solution. High molecular weight

(HMW) forms of HA can have a diameter in the order of 500nm (Laurent 1989).

repeating disaccharide

glucuronic acid Ai-acetylglucosamine

Figure 1.1. Structure of hyaluronan (rnodified from Alberts et al. 1994).

Along the HA molecule there are clusters of adjacent CH groups forming

patches of a highly hydrophobic character. These hydrophobic patches are repeated

at regular intervals on altemate sides of the molecule (Scott 1989). The altemation

of clustered hydrophobic and hydrophilic segments in HA facilitates its interactions

with biological membranes, hydrophobic proteins (e.g.: link protein) and also in HA

self-aggregation (Scott 1989; Scott et al. 1992).

Intramolecular interactions of HA in solution dampen interactions with other

molecules, and promote its neutral space filling and molecule passing roles (Scott

1998). HA networks are more easily formed by HMW HA. Welsh et al. (1980)

showed that an admixture of LMW HA dramatically alter the rheologic properties

of HMW HA which these authors tentatively suggest is an aggregation breaking

effect.

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1.1.2. Family of glysosaminoglycans

HA is one of a family of connective tissue heteropolysaccharides containing

hexosamine, collectively called glycosaminoglycans (GAG). Other prominent

GAGS include heparin sulphate (HS), heparin, chondroitin sulphate (CS), dermatan

sulphate (DS), and keratin sulphate (reviewed in Sarnes 1994; Soldani and

Romagnol i 199 1 ).

HA is the simplest GAG, and is structurally unique in at least three respects.

Firstly it is the only GAG which does not exist covalently linked to a core protein

(Le. as proteoglycan). Secondly it has no sulphate content. Thirdly, its linear

polymers can reach a much greater relative molecular m m . For example, HA in

joint fluid can be up to several million daltons in size, while proteoglycans are

commonly less than 30kDa (Fraser 1994).

1.1.3. Synthesis of HA

Eukaryotic HA synthases are localized to the inner leaflet of the plasma

membrane where they extrude HA directly into the extracellular space (Prehm 1989;

Weigel et al. 1997). HA chains are elongated at the reducing end by alternate

transfer of sugar residues from the substrates UDP-glucuronic acid and UDP-N-

acetylglucosarnine (Prehm 1983a,b). This occurs inside the plasma membrane, and

the chain (with the non-reducing end ahead) is extruded into the pencellular space,

whiIe the growing HA chah remains bound to the enzyme (Prehm 1984; Heldin et

al. 1993). The mechanisrn for release of HA extracellularly is not known. HA

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chah release typically occurs after s20,000 monosaccharides s4 x 1 0 6 ~ a ) have

been assembled. HA extrusion to the extracellular milieu is presently viewed as a

process requiring the enzyme to form a pore or channel-like pocket to guide the

hydrophilic HA chain through the lipid layer (see Figure 1.2). Extrusion of the

growing chain into the extracellular space allows unconstrained growth of the HA

polymer. This mechanism of synthesis and release is in contrast to the smaller

GAGS which are synthesized in the Golgi cornpartment and released by exocytosis

(Hascall et al. 1998).

4 p lasma membrane & .a

UDP- h yaluronan

Figure 1.2. Extrusion mode1 for HA synthesis (modified from Prehm et al. 1983)

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There are three HA synthase (HAS) genes in eukaryotes (Itano and Kimata

1996; Spicer et al. 1996; Fulop et al. 1997; Spicer et al. 1997a). In contrat with

the low levels of identity between HAS within a species (HAS 1/HAS2, 55%

identity; HAS l/HAS3,57% identity; HAS2/HAS3, 7 1 5% identity). the sequence

between individual human and mousc HAS orthologues is highIy conserved

(identity 96%) (Spicer et al. 1997b; Weigel et al. 1997). These sequence

characteristics suggest a conservation of the functionally important residues of HAS

during evoiution and the differences of action of the three HAS proteins (Spicer et

al. 1997b; Weigel et al. 1997). Localization of the three HAS gcnes on different

chromosomes and the appearance of HA throughout the vertebrate class suggest that

this gene family is ancient and the isozymes appeared by duplication early in the

evolution of vertebrates (Spicer et al. 1997b). Recently, Spicer and Nguyen ( 1999)

demonstrated that HAS-2 deficient embryos were deficient in HA and had severe

developmental defects, including a failure to form the endocardial cushions of the

hem (Spicer and Nguyen 1999). HAS- 1 and HAS-3 mutant mice have yet to be

reported.

The HAS protein has four trammembrane domains and one or more

amphipathic helices that may be partially embedded in the membrane (Watanbe et

al. 1996; Spicer et al. 1997a). Translocation of an HA chah across the bilayer by

such a small protein is difficult to envision. It has recenily been shown that

streptococcal HA synthases do not oligomerize, but require cardiolipin molecules

for maximal activity and stability (Tlapak-Simmons et al. 1998). Thus it has been

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proposed that a collection of cardiolipin molecules may function with HAS proteins

to create or maintain a pore-like structure (Tlapak-Simmons et al. 1998).

It seems likely that most cells can synthesize HA at some point in their

natural life cycle and most of al1 during periods of rapid growth (Fraser and Laurent

1989; Fraser et al. 1994). The activity of hyaluronan synthase correlates with a

variety of cellular parameters inciuding ce11 growth, transformation and

differentiation (Prehm et ul. 1980), metastasis (Toole et al. 1979), ceIl migration

(Chen et al. 1989; Schor et al. 1989: Ellis et al. 1992; Ellis et al. 1996) and mitosis

(Moscatelli and Rubin 1975; Brecht et al. 1986). HA synthesis has been proposed

to increase dunng active division and decline at confluence. with fluctuations

during the ce11 cycle peaking at mitosis (Brecht et al. 1986; Matuoka et al. 1987;

Yoneda et al. 1988). HA extrusion ont0 the ce11 surface just prior to mitosis may

create a hydrated microenvironment which promotes partial detachment and

rounding of dividing cells (Brecht et al. 1986; Hall et al. 1994; Koochekpour et ul.

1 995).

HA synthesis is responsive to environmental factors such as hormones

(glucocorticoids, th yroid and sex hormones), vitarnins (vitamin C and retinoic acid).

growth factors (EGF, PDGF, TGFP, FGF, IGF- 1) and cytokines (IL- 1, IL-6 and

TNF) which can directly activate HA synthesis (Heldin et al. 1989; Honda et al.

1989; Sampson et al. 1992; Tammi et al. 1994; Ellis and Schor 1995; Tirone et al.

1997). Many of these factors, especially the growth factors, cm act in autocrine or

paracnne modes, and are frequently related to matrix changes in neoplasia and

inflammation. Furthemore, cyclohexirnide and protein kinase inhibitors have been

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shown to inhibit synthesis, indicating that transcription as well as phosphorylation is

required for activation (Klewes and Prehm 1994).

1.1.4. HA degradation

Circulating levels of HA in human semm are nomally -30-40ng/ml

(Laurent et al. 1995). This is due to a rapid turnover of HA in the bloodstream that

results in a mean half-life for HA of only 2.5 to 5.0 minutes in heaithy adults (Fraser

and Laurent 1989; Laurent et al. 1996). Of the 1 O- 100mg/day entering the

bloodstream the majority (about 90%) goes to the liver, while the remainder goes to

the kidney (Laurent et al. 1987) and spleen (Bentsen et al. 1986).

Studies with separated liver cells (Smedsrod et al. 1984) have shown that

sinusoidal liver endothelial cells are pnmarily responsible for HA uptake.

Metabolic degradation of HA is principally intracellular and relies on receptor

mediated uptake (Eriksson et al. 1983; Smedsrod et al. 1984; Fraser et al. 1985;

Laurent and Fraser 1986; Raja et al. 1988). Endocytic degradation in the liver is

thought to be mediated by HARLEC an HA receptor complex of liver endothelial

cells which has been isolated from rat sinusoidal liver endothelial celis as. This

complex contains three proteins of 100, 166, and 175kDa proteins (Gustafson 1996;

Yannariello-Brown et al. 19961. HARLEC also recognizes chondroitin sulphate

(CS) and dermatan suIphate (DS). Thus CS given intravenously into rats prior to

intravenous HA c m inhibit the uptake of HA by the liver (Gustafson and Bjorkman

1997).

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Abnormal HA accumulation has been noted in a variety of fibrotic disorders

(Whiteside and Buckingham 1989). Examples include sclerodenna (Freitas et al.

1 W6), hypertrophic scarring (Savage and Swann 1985) and fibrosis of liver and

lung (Frebourg et al. 1986; Bjemer et al. 1989). Because HA accumulation in

fibrotic disorden. understanding how HA is removed from connective tissues has

long been of interest (Fraser and Laurent 1989; Roden et al. 1989). Elevation of

serum HA levels also occuts in rheumatoid arthritis, liver cirrhosis and various

malignancies (Engstrom-Laurent 1989: Coppes 1993; Laurent et al. 1996).

HA is turned over locally within cells and tissues, particularly during

ernbryogenesis, tumourigenesis and upon injury. This process occurs in embryonic

tissues during development and also in remodeling tissues (Fraser and Laurent

1989). Numerous ce11 types including human synovial cells (Truppe et ul. 1977),

SV40-trinsformed 3T3 fibroblasts (Culty et al., 1992), embryonic myocardial cells

(Bernanke and Orkin 1984), macrophages (Culty et al. 1992; Gustafson and

Forsberg 199 1) and human breast cancer ce11 Iines (Culty et al. 1994) have been

shown to degrade HA.

Studies both on cultured cells and on developing tissues suggest that the

mechanism of HA degradation may require intemalization/endocytoçis before

degradation. Degradation of HA is thought to occur in lysosomes because the

hyaluronidase is an acidic pH requiring enzyme (Orkin and Toole 1980; Orkin et al.

1982; Bernanke and Orkin 1984). Indeed, inhibitors of lysosomal acidification such

as ammonium chloride and chloroquine block HA degradation (Alston-Smith et al.

1992; Culty et al. 1992).

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HA degradation in macrophages fibroblasts (Culty et al. l992),

chondrocytes (Hua et al. 1993) and several tumour ce11 lines (Culty et al. 1994).

requires CD44 Underhill and coworkers (Underhill et al. 1993; Pavasant et al.

1994) have shown that CD44 mediated removal of HA occurs at several stages of

developrnent. For instance, dunng lung developrnent. macrophages expressing

CD44 increase in number in parallel with a marked decrease in interstitial HA

concentration; injection of a hlocking antibody against CD44 into newbom mice

was shown to cause an increase in lung HA relative to controls (Underhill et al.

1993). In embryonic skin, newly forming hair follicles are associated with

condensing mesoderm; this process of mesodemal condensation is also associated

with lung HA degradation and elevation of CD44 (Underhill et al. 1993). During

bone development, lacunae surrounding hypertrophic chondrocytes are highly

enriched in HA (Pavasant et ni. 1994) and swelling pressure exerted by this HA

contributes to expansion of lacunae as bone growth occurs (Pavasant et al. 1996).

Subsequently, in the zone of erosion, the HA within these lacunae has been

proposed to be removed via CD44-rnediated endocytosis (Pavasant et al. 1994).

Upon injury, metabolism of HA has been observed to rnodify the outcome of a

wound (Burns er al. 1997). Likewise, in neoplasia HA deposition exceeds

degradation (Auvinen et al. 1997; Ropponen et al. 19%).

Under specific conditions HA degradation may also occur extracellularly.

For exarnple, HA within the cartilage ECM becomes partially depolyrnerized, most

likely due to the action of free radicais (Baker et al. 1989; Ng et al. 1992). Free

radicals can be generated by ionizing radiation and are also present at wound sites

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(Forschi et al. I W O ) . Such initial depolyrnerization of HMW HA may an initial

step which facilitates HA catabolisrn. For instance, McGuire et al. ( 1987)

demonstrated that myocardial cells bind, intemalize and degrade partially

depolymerized HA more efficiently than HMW native HA. Thus partial

extracellular together with endocytosis results in the complete degradation of the

HA (Hua et al. 1993).

1.2. Functions of HA

Through its complex interactions with matrix components and cells, HA has

multiple roles in biology. These biological roles range from mechanical functions

in the extracellular matrix to regulation of cellular behavior. HA affects many

biologic properties including wound healing (Chen and Abatangelo 1999),

angiogenesis (Rooney et al. 1995), embryonic morphogenesis (Toole 1997), turnour

metastasis (Bourguignon et al. 1997), inflammation (McKee et al. 1996), cell

adhesion (Hall et al. 1994; Klein et al. 1996) ce11 migration (Chen et al. 1989;

Melrose et al. 1996) and proliferation (Wiig et al. 1996; Bertrand et al. 1997).

1.2.1. Physicochemical functions of HA

HA function has long been attrîbuted to its physical and chemical properties

and its interaction with oiher macromolecules. Because of its water attracting

properties, HA plays a role in the osmotic homeostasis of the extracellular space

(Comper and Laurent 1978; Comper and Zamparo 1990). HA meshworks also

exhibit macromoleular exclusion and thus can regulate the distribution and transport

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of plasma proteins in the tissues such that small molecules move freely in the

network, while larger particles become immobilized (Scott et al. 1989). This steric

exclusion property of HA are thought to serve to protect cells by hindering the

approach of particles that would otherwise cause darnage (Presti and Scott 1989)

and serve as a barrier against the spread of infectious agents (Laurent and Fraser

1992). The rheological properties of HA solutions showing both viscoelasticity and

shear dependence have been related to its lubricating functions in joints and other

tissues (Balzacs and Delinger 1993).

1.2.2. HA binding proteins: the hyaladherins

Although HA is not covalently linked to proteins like other GAGS, several

extracellular intracellular and plasma membrane proteins bind to it with high

affinity (Figure 1.3.). These HA binding proteins, or hyaladherins, are widely

distributed in the body and have diverse functions (Toole 1997). Extracellular and

ceIl surface hyaladhenns are important in modifying matrix assembly, cell-matrix

and ceil-ce11 interactions. Besides their extracellular structural and orgünizational

functions, of particuiar interest is the discovery of hyaladherins that exist within

cells.

Cellular hyaladherins can be grouped into at least two classes of proteins.

CD44 is a prototype of the type 1 transmembrane HA receptor. This protein is

predominantly present at the ce11 surface but can be shed into the extracellular

space. CD44 binds to HA via a complex site known as the link module (Kohda et

al. 1996). RHAMM is a prototype of cell-associated hyaladherins that occur at

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ECM Proteins versicad

CD40 Family hyaluronec tin link protein aggreçan neurocan brevican fibrinogen

Soluble Proteins Trypsin inhibitor 1

I

Figure 1.3. Hyaluronan binding proteins - the Hyaladherins (modified from Pniwand et al. 1999)

multiple cellular loci, including the cell surface, cytoplasm and nucleus. This

group, including RHAMM, cdc37, and p68 are characterized by a lack

transmembrane domain, signal sequence or link module. HA binds to this class of

hyaladherins through short, basic amino acid motifs (Yang er al. 1994; Kohda et al.

1 996).

1.2.2.1. CD44

CD44 was first described as a ce11 surface molecule of T-lymphocytes,

granulocytes, and cortical thymocytes (Dalchau et al. 1980). This protein was then

rediscovered as the phagocytic glycoprotein 1 (Pgp-1) (Mackay et al. 1988), and

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GP90 Hennes (Goldstein et al. 1989). CD44 is now known to be a widely-

expressed receptor for HA (Underhill et al. 1987; Aniffo et al. 1 990).

CD44 consists of an external domain, highly conserved transmernbrane

domains and cytoplasmic domains (Naor et al. 1997). There are 20 known isofoms

of CD44 which are created through alternative splicing of 10 variant exons

(reviewed in Ponta et al. 1998). The smaliest isoform, known as CD44s, lacks al1

ten variant exons. CD44s is the most abundant isoform in vivo. Additional post-

translational modifications of CD44 include phosphorylation and palmitoylation in

the cytoplasmic domain, N- and 0- linked glycosylation and addition of GAGS in

the external domain (Lesley et al. 1993). Dimerization of CD44 has been

implicated in enhanced binding of HA can occur naturally with CD44v4v7

(Sleeman et al. 1997), caii be induced experimentally with certain divalent

antibodies to CD44 (Lesley et al. 1993), or can be induced by constructing CD44

with cysteine in the intramernbranous domain that forms disulfide cross 1 inks

(Perschl et al. 1995).

Interestingly, not al1 CD44-expressing cells are able to bind HA. This

property can be acquired or can occur transiently (Hyman et al. 199 1). The ability

of HA to bind to CD44 is regulated by both protein conformation, rather like

integrin activation, and by glycosylation patterns. Thus, CD44 can be stimulated to

bind HA by phorbol esters, anti-CD44 antibodies, or degIycosylation (Sherman et

al. 1994). Blocking anti-CD44 mAbs studies suggest that topography of the CD44

epitopes and their orientation toward the HA binding site determine the ability of

antibodies to interfere with HA binding (Lesley et al. 1993; Zheng et al. 1995).

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Clustenng of CD44 proteins, which is dependent upon cytoskeletal proteins, also

seems important to its ability to bind HA (Lokeshwar et al. 1994). Certain cells,

including some B and T ce11 lines, appear constituitively able to bind HA.

However. further studies are required to define the molecular mechanisrns that

result in CD44/HA interactions as well as to assess the impact that these

interactions have on ce11 behaviour.

CD44 is a multifunctional receptor involved in cell-ceIl and ceII-ECM

adhesion, i.e. ce11 motility, trafficking, 1 ymph node homing, lymphocyte activation,

presentation of chemokines and growth factors to traveling cells, and transmission

of these growth signals (Lesley et al. 1993). As well, CD44 participates in the

endocytic uptake and intracellular degradation of HA (Culty et al. 1992; Hua et al.

1993), and transmission of signals mediating hematopoiesis and apoptosis (Ayroldi

et al. 1995; Henke et al. 1996) that are relevant to tumour progression and wound

repair. CD44 is constituitively expressed in regions of active ceIl growth (Mackay

et al. 1994).

CD44 expressed on turnour cells and host tissue stroma1 hyaluronan can

enhance growth and invasiveness of certain tumours. Disruption of CD44-HA

interaction by soluble recombinant CD44 has been recently shown to inhibit tumour

formation by lymphoma and melanoma cells transfected with CD44 (Zeng et al.

1998). CD44 is expressed on many types of cancer cells, such as malignant gliomas

(Koochekpour et al. 1995) human breast cancer cells (Culty et al. 1994), colon

cancer cells (Ropponen et al. 1998) ovarian cancer cells (Catterall et al. 1997) and

basal ce11 carcinomas (Baum et al. 1996). A diverse set of CD44 receptor isofoms

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is found on breast cancer ce11 lines, not only among different ce11 lines but also

within individual ce11 lines (Culty et al. 1994). Al1 ce11 lines investigated can bind

HA. Interestingly HA binding and CD44 expression is positively correlated with

invasive potential of breast ovarian and colon cancer ceIl lines (Culty et al. 1994;

Catterall et al. 1997: Ropponen a al. 1998). Disruption of the CD44 function on

mammary carcinoma cells induces apoptosis (Yu et al. 1997). In basal ce11

carcinoma expression of CD44 is found to be down regulated, while the expression

of the CD4.4~6 isoform is increased (Baum et al. 1996). This variant of the receptor

seems to be linked to cellular functions, while the absence of the CD44s accounted

for the noninvasive character of this type of tumour.

The interaction of HA with the CD44 receptor is also involved in

inflammation (Lesley et al. 1997), and liver cirrhosis (Rockey et al. 1998).

Synovial fibroblasts isolated from patients with osteo- or rheumatoid arthritis were

shown to exhibit complex splicing variations in their CD44 expression (Croft et al.

1997). Noninflamed tissue samples showed no such splicing events. It was found

that rheumatoid tissue contained higher levels of expressed CD44, while

osteoarthritic tissue samples showed greater variety in their CD44 expression. The

interaction of HA fragments with the CD44 receptor on macrophages in inflamed

tissues can induce the expression of inflammatory genes of the chemokine family

(IL- 1 B. TNF-a and IGF- 1 ; Noble et al. 1993; McKee et al. 1996) and nitric-oxide

synthase through a nuclear factor kappa beta (NFuP) dependent mechanism (Noble

et al. 1996; McKee et al. 1997). A similar effect of HA fragments on nitric oxide

synthase was observed in liver endothelid cells and Kupffer cells (Rockey et al.

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1998). It is proposed that the various interactions of CD44 with HA fragments

might sustain or increase the process of inflammation.

An extensive body of evidence exists suggesting that CD44 is involved in

angiogenesis (Goshen et al. 1996; Trochon et al. 1996; Deed et al. 1997; Griffioen

et al. 1997; Ozer et al. 1997). Endothelia1 cells of solid turnour vasculature have

been found to display an enhanced expression of CD44, an upregulaiion that could

be induced by angiogenic factors such as FGF and VEGF (Griffioen et al. 1997).

Further, it has been demonstrated that LMW oligosaccharides HA (HAS-50) can

induce angiogenesis (West et al. 1985), while HMW HA in the ECM has been

shown to inhibit angiogenesis (Dvorak et al. 1987; West and Kumar 1989). Both

should bind to ce11 surface recepton but how they have different elfects on cells in

cornparison to HA oligosaccharides is unclear.

CD44 interactions with angiogenic oligosaccharides of HA have also been

found to upregulate the expression of early-response genes such as c-Jos, c-jun,

junB. Krox-20 or Krox-24 in endothelial cells (Deed et al. 1997). The products of

these genes are known to act as transcription factors for genes encoding

extracellular proteinases, which are additional factors that promote endothelial ce11

migration and thus angiogenesis.

1.2.2.2. RHAMM and related proteins

RHAMM is a member of a group of cell-associated hyaladherins including

p68 and cdc37 that localize to several subcellular loci and that perform multiple

functions in regulating ce11 motility and ce11 cycle (Turley et al. 1982, 1994;

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Grammatikakis et al. 1995; Deb and Datta 1996; Mohapatra et al. 1996; Toole et al.

1997; Wang et al. 1998). For instance, cell surface foms of RHAMM are

transiently expressed in most cells but are nevertheless key to regulating cell

motility as determined by antibody blocking experiments (Samuel et al. 1993; Hall

et al. 1994, 1995, Pilarski et al. 1994; Turley et al. 1994; Savani et al. 1995a,

1995b; Nagy et al. 1995; Masellis-Smith et al. 1996; Delpech et al. 1997; Zhang et

al. 1998). Intracellular forms of this class of hyaladherins, including RHAMM,

bind to and chaperone signaling molecules involved in regulating cell cycle and ce11

motility (Grammatikakis et al. 1995; Mohapatra et al. 1996; Kimura et al. 1997).

These types of hyaladherins may also perform functions within the nucleus. Such

hyaladherins typically lack a link module for binding HA but rather utilize short

sequences encoding basic amino acid motifs (Yang et al. 1994) which are required

for ce11 motility and proliferation (Samuel et al. 1993; Sherman et al. 1994). Even

though they are present on the cell surface (Samuel et al. 1993; Sherman et al.

1994; Grammatikakis et al. 1995; Kirnura et al. 1997; Crainie et al. 1999) this class

of hyaladherins is also characterized by an absence of both signal sequences and

transmembrane domains. Therefore, the molecular bais for their subcellular

distribution is not yet clear. Based upon their modular and dynamic subcellular

location and the different mechanisms by which they bind to HA, these proteins

likely regulate cell motility and ce11 cycle in a manner that is fundamentally distinct

from the more well characterized HA receptor, CD44

Surface RHAMM regulates signals generated by both HA (Turley 1982;

Entwistle et al. 1996; Toole et al. 1997) and growth factors such as PDGF (Zhang et

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al. 1998) and TGFP (Samuel et al. 1993). The ability of HA to signal motility via

RHAMM implies that the HA binding domains are also necessary for signal

transduction. Interestingly, the HA binding domains of intracellular foms of

RHAMM are required for activation of erk kinase by mutant active ras and by

growth factors that activate ras, such as PDGF (Zhang et al. 1998).

Signais from HAkell surface RHAMM interactions are associated with

activation of src (Hall et al. 1996), focal adhesion turnover (Hall et al. 1994) and a

reduction in the tyrosine phosphorylation focal adhesion kinase (FAK) (Hall et al.

1995). RHAMM-mediated activation of src is required for HAIRHAMM-regulated

fibroblast motility and both locomotion of these cells and activation of src by HA

can be blocked with anti- RHAMM antibodies (Hall et al. 1996). However, celis

transfected with either a constituitively active f o m of src or v-src no longer require

ceIl surface RHAMM (as detected by antibody blocking) for signaling motility,

indicating that src is downstream of ce11 surface RHAMM. Nevenheless, v-src-

induced disassembIy of focal adhesions cannot occur in the absence of cellular

expression of RHAMM (Hall et al. 1996). This, although not confirmed

experimentally, is most easily interpreted by proposing that intracellular isoforms of

RHAMM are required for src-generated effects on the cytoskeleton and is consistent

with the ability of a RHAMM isofom to CO-immunoprecipitate with src (Hall et al.

1 996).

In addition to its effect on src signding, RHAMM isoforms encoding exon

9, appear under certain conditions (e.g. subconfluence) to control the erk kinase

cascade through ras (Zhang et al. 1998). Mutations of domains of intracellular

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RHAMM isoforms block signaling through ras (Hall et al. 1995) and activation of

erk (Zhang et al. 1998). Specifically, RHAMMA 1-5 (73 kD) interacts specifically

with erk 1 kinase and its upstream activator MEK in transfonned cells (Zhang et al.

1998; Paiwand et al. in preparation) and its overexpression constituitively activates

this cascade. Thus, at least one form of RHAMM, in a manner possibly analogous

to cdc37 (Kimura et al. 1997, Silverstein et al. 1998), directly associates with

kinases that regulate transformation, proliferation and motility (Zhang et al. 1998).

Imponantly, a RHAMM isofortn of the same molecular weight predicted by the

RHAMMA 1-5 cDNA (e.g. 70-73 kDa) is uniquely upregulated after wounding of

smooth muscle ceII monolayea (Savani et al. 1995). Furthemore, manipulation of

RHAMMA 1-5 expression or funciion alters the ability of PDGF, a key growth

factor in response-to-injury processes, to activate signaling cascades (Zhang et al.

1998) and modify actin assembly (Hall et al. 1994, Chang et al. 1997). The role of

erk in these functions remains to be investigated.

A recently characterized intracellular HA binding protein encodes a 29.3kDa

chicken homologue protein homologous to cdc37, an essential ce11 cycle regulatory

factor previously characterized genetically in yeast and Drosophila (Grammatikakis

et ai. 1995). These findings suggest a role for HA in cell division control. Cdc37 is

associated with Cdk4 through HSPSO, further suggesting that cdc37 may regulate

the rnammalian ce11 cycle through direct effect on Cdk4 (and thus the GIS

boundary) (Dai et al. 1996). Further, raf and src are known to directly interact with

HSP90lp50-cdc37 complexes (Perdew et al. 1997).

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A recently characterized 34kDa HABP is a new member of the cellular

hyaladhenns as demonstrated in a wide viuiety of ceIl lines. This protein has been

found to form a homodimer of 68kDa (p68) that binds specifically to HA (Deb and

Datta 1996). P68 has been shown to be highly phosphorylated in transformed

fibroblasts compared to normal fibroblasts, and phosphorylation was enhanced in

the presence of HA, PMA and calyculin-A (Rao et al. 1997). The phosphorylated

form of p68 is show on the ce11 surface and cm be detected in semm free medium

(Rao et al. 1997). P68 has a 100% homology with gClq, the complement protein

(Ghebrehiwet et al. 1994) and has been demonstrated to be the human counterpart

of munne YL2 (Luo et <il. 1994). Thus it has been proposed to modulate the

function of Rev which is expressed at the early stage of HIV 1 and act as viral

transactivator for the replication of HIV I (Das et al. 1997). Interestingly, the cDNA

sequcnce of p68 has complete homology with the cDNA sequence of a protein P-32.

CO-purified with the human pre-mRNA splicing factor SF2 (Krainer et al. 1991).

1.2.2.3. Hyaluronan and tumourigenesis

It has been clear for years that the HA content of tumours is often elevated

(Knudson et al. 1989), which is consistent with the raised concentrations of HA in

semm of cancer patients (Kumar et al. 1989). Although increased HA synthesis is

not a universal characteristic of tumours, there seems to be an overall tendency for

transfomed cells to exhibit higher levels of HA production (Kimata et al. 1983;

Turley and Tretiak 1985).

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In cell culture many cell types have reduced capacities to synthesize HA

even when derived from tumours enriched in HA (reviewed in Knudson et al.

1989). These cm nevertheless stimulate HA synthesis by normal fibroblasts.

Production of tumour associated HA occurs via tumour-stroma1 cell interactions.

Also some human tumour cells also possess unoccupied high affinity ce11 surface

binding sites for HA which rnay allow tumour ceils to interact directly with HA

enriched ECM. This may allow them to use HA as a support for adhesion and

locomotion and guide them into surrounding stroma.

Takeuchi et al. ( 1976) reported that the HA con tent of malignant breast

lesions was higher than that of normal tissue. Bertrand et al. (1992) confirmed the

finding and extended it by showing that the increase was more marked in the

peripheral invasive areas of the tumour than in the central parts. Histochemical

staining localized HA over-expression in the malignant stroma, whereas the tumour

epithelium was HA negative (de la Torre et al. 1993). Further, Auvinen et al.

( 1997) have dernonstrated HA expression was restricted to the stroma1 connective

tissue in benign lesions, while in malignant breast tumours the intensity of stroma1

HA staining was significantly stronger and was also detected in the cell membranes

and cytoplasm of adenocarcinorna cells. Victor et al. ( 1999) recently showed that

the passage of human breast cancer cells from the primary state to the metastatic

state was characterized by a drarnatic increase of HA and hyaluronidase production

and expression of HA-binding sites at the invasion areas. Thus, both synthesis and

degradation of HA presumably support tumour invasion and growth.

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It has recently been shown that invasive breast adenocarcinorna has a

significantly higher level of hyaluronidase activity, correlating with its invasive

potential (Madan et al. 1999). This hyaluronidase can disrupt basement membrane

integrity and produce an angiogenic response that has been implicated in tumour

invasiveness and metastasis (Madan et al. 1999). In contrat, HYALI, a widely

expressed hyaluronidase in somatic tissues (Frost et al. 1997) has been proposed as

a candidate tumour suppressor gene as it is found at the sight of a common tumour

suppressor locus (Csoka et al. 1998). This is a locus frequently deleted either as a

loss of heterozygocity or hornozygous deletion in a nurnber of human rnalignancies,

particularly tobmco-related lesions of the mouth, head and neck, and of the lung

(Killary et al. 1992; Buchhagen 1996). HYAL-I activity is absent or strongly

suppressed in squamous ceIl carcinorna cell lines compared to normal keratinocyte

controls (Frost et al. in prepnration).

In a recent study, HA intensity was shown to be stronger in high grade

colorectal tumours (Ropponen et al. 1998) and to be prognostic of poor outcorne.

The abnormal expression of HA in the neoplastic colon epithelial cells is suggested

to provide a distinct advantage for invasive growth and metastasis.

Itano et al. ( 1999) have recently implicated HA in turnourigenesis through

selection of mouse mammary carcinoma cell lines mutant deficient in HA. These

mutants show loss of HA production and loss of periceIlular HA coats, and more

important, decreased metastatic ability. Further, transfection with the HA synthase

HAS- 1 can rescue these mutants (Itano et al. 1999). In a related study, human

HTlOSO cells were transfected with HAS2, which caused the ceils to grow more

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rapidly and produce tumours that were 2-4 times larger than control (Kosaki et al.

1999), providing more direct evidence for HA'S role in tumourigenicity.

1.2.2.4. Hyaluronan and wound healing

As wound healing progresses in vivo there are changes in the composition of

the extracel1ular matrix. Initially, a wound contains a fibrin-rich matrix, followed

by a HA-nch matrix (Bertolami and Donoff 1978). HA is then removed by

hyaluronidases and then there is a deposition of a fine soft ternporary reticular

collagen, called Type DI, which is in tum repiaced by fiben of Type 1 coilagen. The

latter is the hallmark of a mature scar. In fetal wounds there is no scarring. It is

tempting to speculate that the high concentrations of HA which persist in fetal

wounds (Longaker et al. 1989; 1990; 199 1 ; Mast et al. 1992) may in part underlie

scar free healing.

HA also functions in wound repüir as a regulator of ce11 motility. This may

be mediated through both its physicochemical properties as well as through direct

interactions with cells. in the former, HA appears to provide an open, hydrated

matrix that pemits cell migration (Toole et al. 1997), whereas in the latter, HA:cell

interactions via cell surface hyaiadherins actively promote migration. RHAMM in

particular forms links with several protein kinases associated with ce11 locomotion,

for e.g., ERK, p IZFak and pp6Oc-src (Hall et al. 1994; Hall et al. 1996; Wang et

al. 1998). Kobayashi and Terao (1997) have shown a dose-dependent increase of

the proinflammatory cytokines TNF-a, IL- 1 and IL-8 production by human uterine

fibroblasts at HA concentrations of IOug/ml to lmg/ml via a CD44 rnediated

Page 39: RAPID HYALURONAN UPTAKE ASSOCLATED WITH

mechanism, which may also contribute to ce11 motility as well as impact on the

progress of wound healing.

Consistent with the model that scarless wound healing is due in part to HA.

many reports have attested to the ability of exogenous HA to produce beneficial

wound healing outcome (reviewed in Chen and Abatangelo 1999). In animal

experiments. topically applied HA has been shown to accelerate skin wound healing

in rats (Abatangelo et al. 1983; Cabera et al. 1995) and hamsters (King et al. 199 1 ),

and to enhance repair of tympanic membranes (Hellstrom and Laurent 1987). and

comeal epitheliai wounds (Nakamura et al. 1992). In chronic wounds such as

venous Ieg ulcers, HA application has been shown to promote healing (Falanga et

al. 1996). These results and the observation that intrauterine treatment of fetal

wounds with hyaluronidase convened the healing frorn a fetal type to the scamng

rnodel observed ex utero (Mast et al. 1992; West et al. 1997) are consistent with a

persistence of HA as one necessary feature for scarless healing to take place.

1.3. Rationale of this study

In addition to its role in the ECM and at ceIl surfaces, evidence is growing

that HA is also present in the cytoplasm and nuclei of cells in a number of tissues in

vivo (Margolis et al. 1976; Furukawa and Terayama 1977; 1979; Londono and

Bendayan 1988; Ripellino et al. 1989; Kan 1990; Eggli and Graber 1995).

Furthermore, recent studies have shown that there are a number of intracellular

hyaladherins that may be important in regulation of the ce11 cycle or in gene

transcription (Grarnmatikakis et al. 1995; Deb and Datta 1996; Hofmann et al.

Page 40: RAPID HYALURONAN UPTAKE ASSOCLATED WITH

1998; Zhang et al. 1998). For example, intracellular foms of the hyaluronan

receptor, RHAMM, have been shown to regulate erk kinase activity (Hofmann et al.

1998; Zhang et al. 1998). and a vertebrate homologue of the ce11 cycle control

protein cdc37 was recently cloned and found to bind HA (Grammatikakis et al.

1995). Thus we postulate that intracellular HA may be targeted to sites relevant to

intracellular hyaladherin expression by a novel mechanism, there to exert effects

relevant to diverse cell processes. To investigate this we have created a fluorescent

conjugate of HA which can be used to determine the mechanisrn of transport and

potential functions of intracellular HA.

Page 41: RAPID HYALURONAN UPTAKE ASSOCLATED WITH

MATERIALS AND lMETHODS

Page 42: RAPID HYALURONAN UPTAKE ASSOCLATED WITH

11.1.. Ce11 Culture

Murine IOTU2 parental, ras-transformed (C3) (Egan et al. 1987) or

vRHAMMA 1 4-transfected cells (LR2 I ; Hall et al. 1995); parental and connexin43

transfected gliorna cells (provided by J. Nagy, Winnipeg, MB); and human breast

cancer MDA-MB-23 1 and MCF-7 cell lines purchased from American Type Culture

Coilection (Rockwell, MD) were al1 maintained at 37°C in 5% CO2 on lûûmm

plastic tissue culture dishes (Nunclon) in DMEM (GibcoBRL) supplemented with

10% (vlv) fetal calf semm (FCS) (Intergen) and 1 OmM HEPES (Sigma) (growth

medium), pH 7.3. Cells were routinely subcultured using 0.25% trypsinll mM EDTA

in HBSS (GIBCO BRL) from 80% confluent cultures and passaged at a 1 : 10

dilution. Transfected cells were routinely selected using 900pg/ml geneticin (Gibco

BRL). Al1 cells were used under passage 25. Frozen stocks of cells were prepared

by resuspending the cells in freezing medium (70% DMEM, 20% FCS, 10%

DMSO), and then stored under liquid nitrogen.

11.2. Conjugation of HA with Texas Red

HA (Hyal Pharmaceutical Corp., Mississauga, Ontario) suspended in 20mM

MES (Sigma), pH 4.5, with 30% ethanol, was mixed with three-fold excess EDCI

(Aldrich) as per the number of HA disaccharides. Texas red hydrazide (Molecular

Probes) dissolved in DMF (Aldrich) at a molar ratio of probe to disaccharide of 1 : 10

was added and the mixture was shaken overnight at room temperature.

Unconjugated material was then removed with didysis in 10 000 MWCO

Page 43: RAPID HYALURONAN UPTAKE ASSOCLATED WITH

membranes (Pierce) against 75mM NaCl and 40% ethanol for 6 days at 4°C in the

dark. The product (see Figure 11.1) was lyophilized for storage. Analysis of success

of conjugation was conducted with gel permeation chromatography using refractive

index and UV absorption to detemine purity and the molecular size range of the

bioconjugate (see Appendix 2). The molecular weight of the HA conjugated to the

probe was shown to be lOOkDa and the success of conjugation was shown to be

approximately one rnolecule of Texas Red per fifteen hyaluronan disaccharides (see

Appendix 2 - Figure VII.1.).

TEXAS RED HYDRAZIDE HYALURONAN

Figure II.1. Conjugation of hyaluronan to Texas Red hydrazide

Page 44: RAPID HYALURONAN UPTAKE ASSOCLATED WITH

11.3. Treatment of cells with TR-HA and analysis of resultant

intraceliular fluorescence

Cells were seeded in DMEM (Gibco BRL) supplemented with 10% FCS

(Intergen) at 60% confluence on 25mm sterile g las coverslips (VWR) in 35mm

tissue culture plates (Nuncion) [except cells plated for oligosaccharide treatments,

which were plated on 13mm glas covenlips (VWR) plated in 24 well plates

(Falcon)]. After 8 h at 37°C the culture medium was aspirated, cells were rinsed and

medium was replaced with serum-free DMEM containing 4 pg/ml transfemn

(Human, Gibco BRL) 4pglml insulin (Bovine; Gibco BRL) and lOmM Hepes

(Sigma) (defined medium), for 12 h at 37°C. Where indicated cells were pretrcated

with 5Opghl BrefeldinA (Molecular Probes) for 30 minutes; lOmM colchicine

(Sigma) for 30 minutes; lOOnM phorbol 12-myristate 13-acetate (PMA; Calbiochem)

for up tto 4 h; or 50pg/ml MEK inhibitor (PW98059; Oncogene Science; dissolved

in DMSO; Alessi et ni. 1995) for 2 hours; at 37°C and 5% CO2 in defined media.

The cells were then gently exposed to 100pg/ml (unless concentration otherwise

indicated) TR-HA conjugate in 1 ml of (37OC) defined medium, wrapped in foi1

incubated at 37OC with 5% CO? for 1 O minutes (unless incubation time otherwise

indicated). The cells were then rinsed twice in cold 5X PBS to prevent release of

bound hyaluronate during washing (Underhill and Toole 1980; McGuire et al. 1987),

and fixed in a solution of 2% paraformaidehyde and 1 % cetylpyridinium chloride

(CPC), which precipitaies glycosarninoglycans, in a O. 1 M Na-phosphate buffer pH

7.4 for 10 minutes at room temperature. Coverslips were then washed three times

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for 5 minutes each in 1 XPBS (2.7mM KCI; 1.1 rnM KH2P04; l28mM NaCI; 8.1 mM

Na2HP04; pH 7.4), mounted with elvanol (PVA 15%, glycerin 30%), and viewed on

a Zeiss Axiophot 100 confocat microscope using the 63X objective (unless othenuise

specified). To ensure that confocal images represented internalized TR-HA, some

cultures were exposed ro TR-HA for 10 minutes, washed, then digested with

Streptomyces hyaluronidase ( 1 IU/ml at 37OC for 1 h to remove TR-HA remaining

on the ceIl surface that might interfere with confocal images), prior to viewing.

Images represent a z-axis slice through the center of a cell interior. Analysis was

done with UTHSCA Image Tool version 1.28 (University of Texas Health Sciences

Centre in San Antonio, Texas) using zero thresholding of equal central sections

taken from the perinuclear areas of cells. Staining of cells with different ce11

thicknesses were equilibrated by dividing mean fluorescent intensi ty by the intensity

of the FITC-Dextran counterstain ( 10,ûûûMW lysine fixable; Molecular Probes).

Laser settings were constant for al1 images compared.

11.3.1. Digestion of TR-HA with hyaluronidase

Digestion of 1 5 0 ~ of TR-labeled HA prior to addition to cells was carried

out in acetate buffer pH 5.0, using 15 TRU of Streptomyces hyaluronidase (Sigma)

for 24 h at 37°C in a proteinase inhibitor buffer (25rnglml ovomucoid; 1 .Omg/ml

pepstatin A; 18.6mg/ml iodoacetic acid; 37rng/ml EDTA; 17.1 mg/ml PMSF). The

mixture was heat inactivated at 56°C for 30 minutes to destroy enzyme activity.

Control incubations (minus the hyaluronidase) were carried out under the sarne

conditions. Digestion products were used to treat cells as in IT.3.

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11.3.2. Cornpetition with unlabeled HA and HA oligosaccharides

To assess specificity of uptake. cultures were also exposed to 80pg/ml TR-

labeled HA cornbined with excess (4mg/ml; i.e. 5OX excess) unlabeled HA (Hyal

Pharmaceutical, Mississauga, Ontario) or HA oligosaccharide (HA4, HA8, HA 10,

HA12; provided by R. Tammi, Kuopio, Finland). Cells were treated and fixed as

described above in II.3.

11.3.3. Treatment of cells with Texas Red hydrazide probe alone

Cells were treated with 6.67pg/ml TR-hydrazide probe (equivalent aniount of

probe for 100pg/ml TR-HA given the determined ratio of 1 HA disaccharide per 15

Texas red molecule success of conjugation - see Figure VII.1.) followed by fixation

and analysis as above in II.3.

11.3.4. Treatment of cells with Sytol6

Cells were treated simultaneously with 5p.M Syto 16 live ceIl nucleic acid

stain (Molecular Probes) and with 100pg/rnl TR-HA for 10 minutes at 37°C and

fixed and analyzed as above in 11.3.

11.3.5. TR-HA concentration curves

vRHAMM t -5 transfected, ras transfected and parental 10T 112 rnurine cells,

and MDA-MB-23 1 and MCF-7 breast cancer cells were exposed to 0,O.O 1,0.05,

0.1,0.2,0.5,0.75, 1.25,2.O and 3.0mgM TR-HA, and 1 mg/ml FITC-dextran

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( 10,000MW lysine fixable; Molecular Probes) for 15 minutes at 37OC followed by

fixation and analysis as above in 11.3. Confocal laser settings were kept constant

throughout andysis, on a low, medium and high setting (contrast = 153; 202; 264)

for Texas Red, and on a single laser for FITC-dextran, and different lasers were

equil i brated upon analysis.

11.3.6. TR-HA treatment of cells over tirnecourses

Cells were exposed to 150pg/ml TR-HA for 1 minutes. 15 minutes, 45

minutes, 2 hours, 6 hours and 20 hours at 37°C and 5% CO2. Al1 samples were

incubated with 1 mg/ml FITC-dextran ( 10,000MW lysine fixable; Mol Probes) 10

minutes prior to fixation as above in II.3.

11.3.7. Prebinding of peptides to TR-HA

150pg TR-HA was preincubated with 1 mg/ml of a peptide that mimics the

, Yang et al. 1994) or 1 mg/ml of a HA binding domain 1 of RHAMM (peptidem4''43'*

scrambled peptide of domain 1 which has been shown not to bind to HA (Yang et ai.

1994). for 2 h ai 37OC prior to cell treatment as in II.3.

11.3.8. Anti-CD44 and Anti-RHAMM antibody blocking

Live cells were preincubated for 30 minutes at 37°C and 5% CO? with

5Opg/ml with, anti-CD44 mAbs: KM201 (R&D Systems) or Hennes I (Endogen); or

~ ~ ~ ~ - R H A M M ~ ~ ~ " ~ pAb, or IgG (Sigma) diluted in defined media prior to addition of

SOpg/ml TR-HA for 5 minutes at 37°C and 5% CO?, followed by fixation and

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analysis as in iI.3. (note: KM201 and RH^^^^^ antibodies were dialyzed

exhaustively to rernove sodium azide in 10,000MWCO membranes; Pierce).

11.3.9. AMAC labeled HA-oligosaccharide treatment of cells

Cells were treated with 1.67mg/ml AMAC Iabeled hyaluronan

oligosaccharides of discrete length (HA& HA12, HA26, HA30; provided by R.

Tammi, Kuopio, Finland), for 10 minutes at 37OC, and fixed and analyzed for

fluorescence as described in iI.3.

11.4. Wounding assays

100% confluent 10T 1/2 ce11 monolayers growing in DMEM (Gibco BRL)

supplemented with 10% FCS (Intergen) were scratch wounded, rinsed w i th fresh

DMEM/! 0%FCS and allowed to recover for up to 24 hours before TR-HA uptake

and analysis was performed as in II.3. Trypan blue (Gibco BRL) addition was used

to exclude areas of cells which sustained unrepaired injury.

11.5. Immunofluorescence

10T1/2 cells were seeded at 70% confluence, ont0 25mm glas coversiips and

allowed to attach for 24 hours in growth medium, washed and placed in defined

medium for 24 hours. Cells were rinsed 5x in lXPBS (pH 7.3) and fixed with

freshly prepared 3 8 paraformaldehyde (Sigma) in PBS, for 10 minutes at RT,

washed 5x for 5 minutes with LXPBS. Cells were permeabilized for 2 minutes with

0.2% Triton X-100 (VWK) in PBS, and washed 5 x for 5 minutes with 1XPBS. Cells

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were then blocked for 1 hour at RT in 3% BSA (Sigma) in PBS washed and

incubated with 1 OAbs: anti-RHAMM R 10.1 pAb or anti-caveolin mAb

(Transduction Laboratories) at 150 in 1 % BSA (Sigma) in PBS, at 37°C for 1 hour.

Cells were then washed 5x for 5 minutes in lXPBS and incubated with 2"

antibodies: goat anti-mouse-RITC (Jackson) and goat anti-rabbit-FITC (Jackson) for

1 hour at 37OC in the dark, then washed 5x in IXPBS. Coverslips were then washed

in xylene, dried on paper towels, and mounted using "anti-fade" media (l0mM p-

phenylenediamine, l 18mM Tris-HCl, 90% glycerol, pH 7.4) and sealed. Cells were

visualized with a Nikon confocal laser scanning microscope.

11.6. Flow cytometry (FACS analysis)

70% confluent IOT1/2 cells, which had been in defined medium for 24 hours

previously, were incubated with 2.0m.M EDTA (ethylene-diaminetetraacetic acid) in

HBSS (Hank's balanced salt solution, Gibco), with 1OmM Hepes, for 5-6 minutes,

gently harvested with FACS buffer (10% FCS in HBSS with 20mM Hepes), and

pelleted by centrifugation at 1500rpm for IO minutes. Cells were then resuspended in

FACS buffer. An aliquot of the ce11 suspension was stained with trypan blue (0.4%

Sigma) and ceilular viability was determined to be above 90% using a

hemacytometer and trypan blue exclusion. Samples of 3x 10' cells were treated in

suspension with O. 1Wml HA (Healon) over a time course of 0,0.5, 1,3,5, 10, 15,

20, 25,30,60 minutes, followed by wash and centrifugation at 4OC, and

resuspension in 0.5ml of wash solution (HBSS; 10% FCS; lOmM Hepes) at 4°C.

Cells were then incubated with 1 : 100 dilution of 1 O Ab: anti-RHAMM R 10.1 pAb,

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or IgG alone, for 45 minutes at 4OC, washed by centrifugation 2x, and resuspended

in wash solution. Cells were then incubated with 2' Ab: Goat anti-RHAMM-cy3 for

45 minutes at 4"C, washed 2x, and resuspended in 1 % paraformaldehyde (Sigma).

Cells were scanned using a coulter electronics EPICS 754 flow cytomeier for surface

RHAMM expression. Al1 tests were duplicated 3x.

11.7. DNaseI digestions

Genomic DNA was isolated from ras-transformed ceIl monolayers (as shown

in Current Protocols). Briefly cells were suspended with trypsin (Sigma), washed 2x

in HBSS, and resuspended in lysis buffer with proteinase K and incubated with

shaking at 50°C for 18 hours, then extracted. Precipitate was recovered with g las

rod and resuspended in lOmM Tris pH 8.0. Extraction was repeated with RNase

treatment.

Each sample consisted of: 0.24pg genomic DNA, 5p1 of 2x DNA binding

buffer and either 2 . 5 ~ 1 ( IOpg or 50pg) of HA or chondroitin sulfate or water.

Samples were incubated together at RT for 2 hours with shaking. Then DNaseI (0.0,

0.6, 1.8. 3.0,4.2, 5.4,6.6, or 7.8 x 104units respectively) was added to each sarnple

and incubated for a further 30 minutes. DNaseI was inactivated by heating at 65°C

for 15 minutes with 5mM EDTA. Sarnples were loaded onto a 1% agarose gel.

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CHAPTER III

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111.1. Texas Red labeled hyaluronan (TR-HA) is rapidly taken up

into ras-transformed cells and targeted to cell processes and the

nucleus.

Optical sectioning of cells, from which cell surface-bound TR-HA has been

removed with hyaluronidase, shows an intracellular location of the TR-HA within 10

minutes after treatment of subconfluent monolayers of ras-transfomed cells (Fipre

111.1.). TR-HA rapidly accumulates within ce11 lamellae and nuclei (Figure III.1.).

Some general cytoplasmic and striking perinuclear accumulation is also observed

(Figure 111.1.). TR-HA within the nucleus is seen in the same confocal plane as a

live cell nucleic acid stain. confirming its location in the nucleus (Figure 111.2.)

Streptomyces hyaluronidase-digested TR-HA (Figure I113a.) or TR-HA rnixed with

excess unlabeled HA (Figure I11.3b.) block of the TR-HA uptake by cells,

indicating that unlabeled HA cornpetes effectively for the uptake of TR-HA and that

the uptake requires larger than hexa- saccharide units of HA respectively. Cells

treated with Texas-red hydrazide aione (Figure Il1.3c.) show no staining.

111.2. TR-HA uptake is dose and time dependent and increases with

cellular transformation.

TR-HA concentration curves indicate that uptake of TR-HA is dose

dependent until an apparent saturation point is reached (Figure III.4.A.). Of the

five ce11 lines examined, the highest levels of intracellular HA accumulation occur

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in ras and RHAMM transformed and as compared to their parental IOT112 cells

(Figure 1IIA.A.). Further, overd1 HA uptake is enhanced in invasive human breast

epi thelial ce11 line MDA-MB-23 1 cells as compared to the non-invasive MCF-7

breast cancer cell lines (Figure II1.4.A.) (note, MDA-23 I contains mutant active

ras, while MCF-7 does not). When these ce11 lines are examined over a 20 hour

tirnecourse (Figure 111.4.B.) it is seen that TR-HA uptake achieves its highest levels

within the first hour of incubation and decreases thereafter. Confocal images

illustrate differences in both TR-HA intensity and intracellular distribution amongst

the five cell lines (Figure III.4.C.). RHAMM and ras transformed, as well as

MDA-23 1 invasive breast cancer cells accumulate significant amounts of

intracellular TR-HA, funher, HA appears to be targeted strongly to the tips of ceil

processes and lamellae of these cells (Figure III.4.C.). Interestingly, non-

transformed IOT1/2 and non-invasive MCF-7 (which do not express mutant active

ras) cell lines display weaker TR-HA accumulation and more diffuse intracellular

distributions.

111.3. Specific HA oligosaccharides inhibit TR-HA uptake

Simultaneous treatment of cells with TR-HA and excess amounts of different

sizes of HA-oligosaccharides were used to determine the minimum effective length

of oligosaccharide which would interfere with cellular uptake of the TR-HA probe.

Cells treated with HA oligosaccharides composed of ten monosaccharides (HA IO) or

fewer appear to have no significant effect upon TR-HA uptake (Figure In.5.).

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However, cells treated wi th excess HA oligosaccharide of 1 2 monosaccharides

(HA 1 2) show a significant overall decrease in TR-HA intracellular intensity (Figure

III-S.), indicating that the HA 12 is the minimum effective length of oligosaccharide

that is able to effectively compete with full length TR-HA for uptake. However the

degree of blocking with HA12 does not achieve the level of inhibition shown for

cornpetition with excess unlabeled HA (Figure 1II.S.A.g).

111.4. Thirty HA-saccharide minimum length is required for

effective targeting of HA to the nucleus.

In order to determine the minimum size of HA necessary to achieve cellular

uptake and intracellular targeting to different cellular compartments. different sizes

of HA oligosaccharides were fluorescently labeled such that their intracellular

accumulation could be monitored. It appears that HA8 and HA12 are internalized by

celis at extremely low levels, if at d l , while HA26 is intemalized and intracellularly

distributed into discrete perinuclear vesicles. Interestingly, HA30 is internalized and

distributed into both perinuclear structures and to the nucleus (Figure 111.6). Thus it

would appear that a minimum of 30 HA monosaccharides is necessary for nuclear

targeting.

Ili.5. Anti-CD44 antibodies affect TR-HA uptake

The KM-201 mAb blocks the binding of HA to CD44 in murine (Culty et al.

1992) hamster (Miyake et al. 1990) and human cells (Thomas et al. 1992). TR-HA

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uptake into cells significantly decreases in both RHAMM transfected, and 10T 1/2

cells following preincubation with KM201 mAb (Figure III.7.A.B.).

111.6. Subcellular distribution of RHAMM is dramatically altered

upon HA treatment.

Immunofluorescent staining of semm starved 10T 1/2 cells shows that

RHAMM and caveolin CO-localize (Figure III.8a.). Within 5 minutes after HA

addition RHAMM leaves the caveolin compartment and the two no longer colocalize

(Figure 111.8.b.). Further. flow cytometry shows that HA induces a rapid fluctuation

in display of ce11 surface RHAMM expression during the first 15 minutes of HA

treatment.

111.7. RHAMM peptides block TR-HA targeting to the nucleus.

Preincubation of TR-HA with peptides that mimic the HA binding domains

of RHAMM (Yang et al. 1994) causes a dramatic decrease in the degree of nuclear

accumulation of TR-HA, while targeting of TR-HA to the cytosol, perinuclear area

and ceIl processes appears unaffected (Figure III.9.a.). This effect appears to

require the HA binding capability of the peptide, since pretreatment of TR-HA with

scrambied peptides made of these domains which lack the ability to bind HA (Yang

et al. 1994) will target HA to $1 intracellular compartments including the nucleus

(Figure II1.9.b.).

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111.8. TR-HA uptake is unaffected by blocking with RHAMMexon9

antibody . In an attempt to determine the roie of RHAMM in TR-HA upiake we

preincubated cells with anti RHAMMexon9 pAb pnor to treatment with TR-HA.

Intracellular accumulation and subcellular targeting within RHAMMAI-5 and

parental IOT112 cell cell lines was unaffected by the presence of the antibody

(Figure 111.10.).

111.9. HA uptake varies depending upon length of ce11 plating time

and posi tively correlates wi th functional cellular RHAMM levels.

Image analysis of relative cellular intensities indicates that when cells are

subcultured and allowed to attach for only 3 hours, RHAMM transfected and

parental 1 OT 1 /2 cells appear to take up TR-HA equally. but by 1 2 hours after

attachment the capability of RHAMM transfected cells to take up HA significantly

exceeds their parental 1 OT 1 /2 counterparts (Figure 111.11.). 24 houn after

subculture RHAMM transfected cells are still maintaining their high levels of TR-

HA uptake, while parental IOT1/2 cells have dropped to IR their original intensity -

the same basal level at which another set of WAMM transfectants, those with

rnutated HA binding domains, have been displaying intracellular TR-HA uptake at

al1 time points (Figure 111.11.).

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III.10. Connexin43 transfected cells show increased TR-HA uptake

and targeting to the tips of the ce11 processes.

Treatment of connexin43 transfected glioma cells with TR-HA results in

cellular uptake to the cytoplasm, nucleus and notably to the tips of the ce11 processes

(Figure III.lZ.a.), whereas parental glioma cells do not target HA to the tips of ce11

processes (Fipre II1.12.b.). When these same cells are plated at confluence and

treated with TR-HA it is seen that intracellular TR-HA intensity is much higher in

connexin43 transfected glioma cells (Figure III.12.c.), than in their parental glioma

counierparts (Figure III.12.d.).

111.11. TR-HA uptake is decreased upon MEK inhibition

Treatment of breast cancer cells with a specific inhibitor for MEK

(PD098059; dissolved in DMSO; Alessi et al. 1995) prior to TR-HA treatment show

that TR-HA uptake is dramatically reduced upon MEK inhibition (Figure III.13.A.

a-c). as compared to cells treated with DMSO alone (Figure II1.13.A. d-f'). Image

analysis indicates that reduction in fluorescent intensities is significant (Figure

III.13.B.).

III.12. HA is targeted to the ce11 processes upon phorbol ester

treatment of parental 10T1/2 cells.

1 OT 1 /2 cells treated wiih TR-HA for 10 minutes show diffuse cytoplasmic

and nuclear staining (Figure 111.14.a.). Following 45 minutes with phorbol ester

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1 OT1/2 cells exhibit an intracellular accumulation pattern of TR-HA in the cytoplasm

that resembles ras-transfomed cells in that TR-HA accumulation occurs at the tips

of ce11 processes (Figure I11.14.b.). Furthemore, treatment of cells with PMA for 4

h appears to enhance TR-HA uptake in a dramatic cytoplasmic and lamellar pattern

(Figure III.14.c.).

IIIml3m TR-HA shows enhanced uptake and targeting to the tips of

the ce11 processes at the wound edge in parental 10T112 cells.

Scratch wounding of 10T1/2 cell monolayers shows that TR-HA

accumulation is enhanced at the wound edge and that celis at the wound edge show

HA targeting to the tips of ce11 processes (Figure III.15.A.B.). Cornparison of cells

30 minutes and 24 hours post wounding indicates that TR-HA uptake enhancement

is most pronounced immediately after wounding and declines with time (Figure

111.15.C.).

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FigureIII.1. Confocal optical sectioning of ras-transformed cells exposed to

100pg/rnl TR-HA. Consecutive optical sections (beginning from the basal surface of

the ce11 and proceeding ai 0Spm increments in the z-axis towards the apical surface

of the cell) show TR-HA in the cell processes (indicated by arrow), perinuclear area

and nucleus of a ras-transformed 10T1/2 fibroblast 10 minutes after its addition to

the culture medium. Bar = IO Pm.

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Figure 111.2. Cells treated simultaneously with 100pg/ml TR-HA and 5p.M Syto 16

live cell nucleic acid stain for 10 minutes at 37°C. (a) Dual fluorescence image of

Syto16 and TR-HA shows staining of the two probes in the same z-axis plane of

confocal imaging, though there is no signal colocalization. (b) TR-HA image of a.

shows fluorescence in the nucleus and perinuclearly. ( c ) Syto-16 image of a. shows

fluorescence in the nucleus. (d) Phase contrast image of the cell. Bar = 5pm.

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Figure 111.3. (a) 100pg/ml TR-HA digested with Streptomyces hyaluronidase prior

to its addition to cells, (b) 100pg/ml TR-HA incubated simultaneously with 4mg/ml

unlabeled HA. Both treatments abolish cellular uptake of TR-HA. (c) Cells treated

with an equivalent amount of TR-hydrazide probe show no staining. Ali incubations

were for 15 minutes at 37°C Bar =lOpm.

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Figure 111.4. A. The amount of uptake of TR-HA by cells is proporticnal to the

concentration of TR-HA applied, until an apparent saturation point is reached (at

-2mg/ml), at which the amount of uptake does not increase any funher. Five cell

lines are shown here, and it appears that ras and RHAMM transformed ce11 lines

intemalize HA to a greater extent than their parental IOT 112 counterparts. As well.

HA uptake is enhanced in invasive MDA-MB-23 1 breast cancer cells compared to

their non-invasive MCF-7 counterparts. n=SO. B. After an initial peak in intensity at

- 1 hr of TR-HA incubation, levels of internalized HA decline over a -20hr

timecourse. RHAMMA 1-5 transfected fibroblasts display the greatest HA uptake

over the timecourse. n=50. C. Confocal images of the five celi lines used in A and B

show (a) RHAMMA 1-5 transfected 10T 1/2 fibroblasts (b) ras-transfected 10T 112

fibroblasts (c) parental IOT112 fibroblasts (d) MCF-7 human breast cancer cells (e)

MDA-23 1 human breast cancer cells; treated with 10pg/ml TR-HA for 15 minutes at

37°C. (a) Ras and (b) RHAMM transfected fibroblasts and (e) MDA-23 1 human

breast cancer cells show TR-HA accumulation in ce11 processes, nucleus and a

str i king amount perinuclearly. (c) Parental 10T 112 fibroblasts show TR-HA

accumulation diffusely in the nucleus and in the cytoplasm. (d) MCF-7 cells show

weak TR-HA signal across the entire cell. Bar = 1Op.rn.

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,, 4 ras-transfected

E l-~ RHAMM-transfected -14-- 10T1/2 parental

m w?- MCF-7 (non-inmsiw) ..-.--.--*._.._ -a- - . MDA-MB-231 ( insiw) *-•

m c

0.0 0.5 1 .O 1.5 2.0 2.5 3.0

TRHA treatment concentration (mglml)

-i-- parental 1 OT1/2 4 ras-transfected +, RHAMM-transfected

- ,I - - MCF-7 (non-inasiw) - -e - . MDA-231 (inasim)

O 2 4 6 8 10 12 14 16 18 20

time of TRHA incubation (hourr)

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Figure 111.5. A. Confocal images of RHAMM transfected cells treated with 4rng/ml

excess HA-oligosaccharide: (a) none, (b) HA4, (c) HA6, (d) HA8, (e) HA 10, (0

HA 12, or (g) 100kDa HA; at the time of TR-HA addition show an overall decrease

in TR-HA intracellular intensity in treatments which included oligosaccharides

composed of (f) 12 saccharide units, though inhibition does not achieve the level of

inhibition obtained using (g) full length (1ûûkD) unlabeled HA. Bar = IOpm. B.

Analysis of cellular intensities in A. n=30. Bars represent SEM.

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Figure 111.6. A. RHAMM transfected cells treated for 10 minutes at 37°C with

1.67mg/ml AMAC labeled oligosaccharides of (a) HA8, (b) HA12, (c) HA26, and

(d) HA30 (monosaccharide units of HA). HA8 and HA12 do not appear to be taken

up into cells to any significant degree. HA26 is intracellularly distributed into

discrete perinuclear vesicular structures, whereas HA30 appears to be the minimum

effective size of HA that shows targeting to the nucleus. (a'-d') show phase-contrat

images of the cells. Bar = 1Op-n. B. Image analysis of (n=4O) cells shows mean

fluorescent intensity of n=40 cells of each sample. Bars indicate SEM. Line indicates

degree of autofluorescence in this experiment.

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Figure 111.7. A. Preincubation of (a) RHAMM transfected fibroblasts and (b)

parental 10T112 fibroblasts with anti-murineCD44 mAb KM201 (50pg/ml), which

has been previously shown to block HA binding to CD44 (Culty et al. 1992; Hua et

ui. i993), appears to significantly decrease TR-HA intemalization in both ce11 lines

as compared to preincubation of (c) RHAMM transfected fibroblasts and (d) parental

IOT 112 fibroblasts with IgG. B. Image analysis of experiments in A. n=60.

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RHAMM transfected parental 1 OT1/2

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Figure 111.8. (a) 10T112 cells serum starved for 24 hours show colocalizirtion

( yellow ) between caveolin (green fluorescence) and RHAMM (red fluorescence).

(b) Addition of HA (O. lpg/ml) for 5 minutes caused a redistribution of RHAMM,

and a reduction of its colocalization with RHAMM over the ce11 body. Bars = 1 Opm.

(c) Flow cytometry of 10T112 cells serum starved 24 hours prior to treatment with

HA (O. lpg/ml) for up to I hour, and stained for RHAMM show that ce11 surface

RHAMM intensity fluctuates rapidly within the fint 15 minutes after HA addition.

Blocking with RHAMM fusion peptide could ablate fluctuation (data not shown)

Error bars = SEM.

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O 10 a0 30 43 90 €D

10ndml HA incubation (minutes)

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Figure 111.9. RHAMMAI -5 transfected cells treated for 10 minutes with 100pg/ml

TR-HA that has been preincubated for 2 hours with: (a) 1 mglml of a peptide

mimicking an HA binding domain of RHAMM will traffic TR-HA to the ce11

processes and perinuclear area but no TR-HA signal is evident within the nucleus:

(b) I mglm1 of a scrambled peptide that is unable to bind HA show distribution of

HA to al1 intracellular compartments. Arrows indicate ce11 processes. Bar = 10pm. n

= 50.

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Figure 111.10. A. Confocal images of parental (a, c, and e) and RHAMM

transformed (b, d, and f) 10T1/2 fibroblasts preincubated for 30 minutes with: (a+b)

Opg/ml, (c+d) I O~glml and (e+D 50ug/mI, of anti-RHAMM an tibody pnor to TR-

HA incubation. Intracellular accumulation of TR-HA appears unaffected by

antibody treatments. Bars =25pm. B. Image analysis of cellular intensities of A.

(n=60). Error bars indicate SEM.

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Figure IILl1. RHAMMA 1-5 transfected, mutant RHAMMA 1-5 (unable to bind

HA) transfected, and parental 1 OT 112 fïbroblasts were subcultured and allowed to

attach for 2, 12 or 24 hours prior to TR-HA treatment (150pglml; 10 minutes). Cells

were analyzed for fluorescent uptake of TR-HA. While RHAMMAI -5 transfected

and parental IOT112 levels of uptake are initially equal. by 12 hours after plating

RHAMMA 1-5 transfectants clearly have more intracellular TR-HA. 24 houn after

plating, parental 1OT 112 levels of uptake decrease until they are equal to HA uptake

of the transfectants mutated in their HA binding domains, which have had

significantly lower TR-HA intracellular staining at al1 three plating times. n=60.

Error bars represent SEM.

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RHAMMA 1-5

O parental 1 OT112

RHAMMAI-5 HABD- T

2 hours 12 hours 24 hours

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Figure 111.12. TR-HA ( 1 ûûpg/ml; 10 minutes) treated (a) connexin43 transfected

glioma cells display nuclear and cytoplasmic HA accumulation as well as high levels

of TR-HA targeted to the tips of the ce11 processes, while (b) parental glioma cells do

not show HA targeted to the tips of cell processes. (c) These same connexin43

transfected cells plated at confluence show greater overall TR-HA uptake than their

(d) parental glioma counterparts plated at confluence. Bars = IOpm.

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O DMSO

PD98059

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Figure 111.13. A. Human breast cancer cell lines: (a,d) MDA-MB-23 1 , (b,e) MCF-7

and (c,f) MCF- IO neo 2T treated (a-c) with 50p.M MEK inhibitor PD098059 or

DMSO alone (d-f) for 2 hours prior to treatment with TR-HA ( lûûyg/ml; 10

minutes). Bar=lOpm. B. Analysis of mean fluorescent ce11 intensities indicates that

al1 three ceIl lines display reduced TR-HA uptake in the presence of MEK inhibitor.

Bars represent SEM. n=30.

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Figure 111.14. Confocal analysis of (a) 1 OT ln parental fibroblasts treated with TR-

HA (100pg/ml; 10 minutes) shows TR-HA distributed to the nucleus and diffusely

present in the cytoplasm. (b) 10T112 fibroblasts treated with 1 OOnM PMA for 45

minutes prior to TR-HA treatment show TR-HA localized at the edge of cell

processes and increased in the perinuclear area, although nuclear stüining remains

unchanged. (c) lOT112 fibroblasts treated with lOOnM PMA for 4 h pnor to TR-HA

addition show dramatically enhanced uptake of TR-HA into ce11 processes. Bar =

10pm.

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Figure 111.15. A. 1 OT 1 /2 ce11 monolayer scratch wounded and treated with TR-HA

30 minutes post wounding shows TR-HA strongly in the nucleus, perinuclear area

and ce11 processes of cells at the wound edge. Those cells that are further from the

wound edge display much weaker TR-HA staining. Bright outline indicates cells at

the wound edge. Arrows indicate ce11 processes. Image captured with 63x objective.

B. Analysis of ce11 intensities in different layers of the 30 minute post-wounded

culture indicates that TR-HA uptake significantly increases with proximity to the

wound edge. n=30. Bars indicate SEM. C. Confocal images captured with the 40x

objective show the wound edge of 10T 112 parental fibroblast cultures. While

enhanced uptake at the wound edge can be seen clearly at (a) 30 minutes post

wounding, the contrast is less vivid (b) at 24 hours post wounding, though cells at

the wound edge do clearly demonstrate increased intracellular TR-HA.

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DISCUSSION

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Whereas intracellular HA degradation in the lysosomes has been extensively

reviewed in many studies (Truppe et al. 1977; Orkin and Toole 1980; Orkin et al.

1982; Bemanke and Orkin 1984; Alston-Smith et al. 1992; Culty et al. 1992; Hua et

al. 1993; Culty et al. 1994). few studies have focussed upon the modes by which

hyaluronan may target to altemate intracellular sites (Collis et al. 1998; Evanko and

Wight in press). This is interesting, since hyaluronan has been localized to many

such altemate subcellular sites including the nucleus (Margolis et al. 1976;

Furukawa and Terayama 1977; 1979; Castejon and Castejon 1987; Ripellino et al.

1988; Kan 1990: Eggli and Graber 1995: Evanko and Wight in press), rER and golgi

(Kan 1 WO), caveolae (Eggli and Graber 1993, mitochondria (Kan 1990; Eggli and

Graber 1995) and membrane mffles (Evanko and Wight in press). Since the HA

synthases are present only at the ce11 membrane and are thought to extrude newly

synthesized HA directly into the extracellular space (Prehm et al. 1989; Weigel et al.

1997). presumably mechanisms must exist to deliver HA back into these intracellular

compartments. These intracellular sites have also been shown to contain

intracellular hyaluronan binding proteins which have a number of significant roles in

ce11 signaling. We begin to delineate some aspects of a novel HA intemalization

process, and its cellular mechanisms in this study.

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IV.1. The phenornenon of rapid HA uptake

Fluorochrome-tagged HA can be rapidly taken up by the transfomied ce11 and

accumulate wi thin subcellular compartrnents, such as ce11 processes (Figure 111.1)

and the nucleus (Figure III.2), which have been shown to contain intracellula-

hyaladherins (Frost er al. 1990; Grammatikakis et al. 1995; Deb and Datta 1996;

Entwistle et al. 1996: Zhang et al. 1998). The TR-HA uptake process is HA specific

since it is blocked by predigestion of the TR-HA probe with hyaluronidases (Figure

II1.3.a.). is effectively competed by excess unlabeled HA (Figure 111.3.b.), and since

TR hydrazide alone is not taken up by cells (Figure II1.3.c.).

The presence of nuclear HA is consistent with histological studies in which

endogenous HA has been localized to nucleoli, nuclear clefts, heterochromatin and to

areas of condensed chromatin in the nuclear periphery of oocytes, cumulus cells, and

smooth muscle cells using nanogold labeled hyaladherins (Kan 1990; Eggli and

Graber 1995; Evanko and Wight in press) and in biochemical studies of rat liver and

brain nuclei purified by cell fractionaiion (Margolis et al. 1976; Purukawa and

Terayarna 1977; 1979). Our observations that exogenous HA can rapidly target to

the nuclear cornpartment are supported by laser scanning confocal microscopy,

which resolves the fluorescent signal in adjacent 1 .Opm thick optical sections

(Figure III.l.), and demonstrates that TR-HA staining is present in the sarne focal

plane as a nucleic acid specific stain, confirming its nuclear localization (Figure

111.2.).

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The process appears to be dose dependent and saturable in at least five cell

Iines as demonstrated by concentration curves (Figure III.4.A.). This indicates that

it is dependent upon some limiting factor, such as receptor sites. Examination of

TR-HA uptake over a 20 hour tirnecourse demonstrates the rapidness of this process,

as the greatest arnounts of intracellular HA are seen within 1 hour of TR-HA

incubation (Figure 111.4.B.).

IV.2. Mechanisms of HA uptake

IV.2.1. The potential role of cell surface receptors in TR-HA uptake

TR-HA uptake was decreased in the presence of excess HA oligosaccharides

of 12 monosaccharides in length (HA 12) (Figure 111.5.). It remains unclear whether

HA 12 is able to enter the ce11 itself (Figure III.6.), and thus HA 12 may be

interfering with TR-HA uptake through direct cornpetition through cellular

internalization (and consequent involvement of intracellular sites) as full length

unlabeled HA does, or by statically occupying binding sites in the HA uptake

machinery at the cell surface. One candidate for HA 12 binding is CD44, since it has

been showed previously to require a minimum binding site of HA6 (Culty et al.

1992; Hua et al. 1993). Tammi et al. (1998) have more recently showed that

displacement of HA pericellular matrices frorn CD44 in vivo requires HA l O and they

postulate that tight HA binding necessitates dimerization of CD44 or its cooperative

interaction with other cell surface molecules. Our result of HA12 being required to

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inhibit intemalization of HA (Figure 1115.) is consistent with a cornplex process

potentially mediated by aggregates of CD44, or the involvement multiple forms of

ce11 surface receptors. The inhibition of TR-HA achieved using HA 12 was

incomplete, as evidenced by the rnuch greater degree of blocking achieved using

unlabelled HA (Figure 111.5.). Thus, a significant amount of HA uptake is still

occurring in the presence of HA1 2. It is likely that larger oligos would be more

effective at blocking HA internalization, further suggesting the complexity of the ceIl

surface/ intracellular protein binding interactions at work here.

Interestingly, it appears that nuclear targeting of HA requires a minimum of

30 monosaccharides (Figure 111.6.). Since no single protein has been shown to

require such a large number of HA saccharides for binding it would appear that if

protein binding is important for nuclear entry (as implicated by Figure II1.9.), then

either a complex of proteins, or a novel hyaladhenn is required for nuclear

trafficking of HA.

HA uptake to the lysosomal degradation pathway has been shown to be

mediated by a clathrin dependent process (Alston-Smith et al. 1992) which involves

CD44 (Culty et al. 1992; Hua et al. 1993), as determined by antibody blocking. In

this study we treated cells with two different anti-CD44 antibodies prior to exposure

to exogenous TR-HA. It would appear that CD44 plays a role in the rapid uptake of

HA into 10T1/2 cells, since the KM201 antibody, which has previously been shown

to block CD44-HA binding (Culty et al. 1992; Thompson et al. 1992; Hua et al.

1993), was able to significantly inhibit TR-HA uptake in both transformed and non-

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transformed cells (Figure 111.7.). It should be noted that the block was not a

complete one, which is consistent with the involvement of other molecules. It would

appear that CD44 is involved in targeting HA to multiple subcellular compartments

independent of the previously reported degradative pathway.

A role for the another HA receptor, RHAMM, is also suggested by drarnatic

alterations of RHAMM cytoplasmic and cell surface distributions upon HA

treatment (Figure 111.8.) and by the ability of RHAMM peptides to block HA uptake

to the nucleus (Figure 111.9). The redistribution of RHAMM to the ce11 periphery,

and indeed to the ce11 surface (Figure 111.8.~) as shown by flow cytometry, upon

exogenous HA administration however, is interesting, because it occurs rapidly, as

does HA uptake. Ul trastructural analyses have demonstrated that HA is associated

with caveolae (Eggli and Graber 1995), and these results would collectively

implicate pinocytosis as a potential mode of TR-HA uptake wonhy of further study.

CD44 is potentiafly found associated with the caveolae as well, since CD44 has been

demonstrated to have a strong association with GPI domains (llangumaran et al.

1997). Further, RHAMM and CD44 have been shown to colocalize and

coimmunoprecipitate in human breast cancer cells (Paiwand et ai., in preparation),

and could act cooperatively to mediate HA uptake.

Although RHAMM HA-binding peptides blocked nuclear uptake of TR-HA

(Figure III.9.), pretreatment of cells with anti-RHAMM antibodies proved

inconclusive (Figure II1.10.). These results suggest that the HA binding property

and not other domains shown to be necessary for cell motility, which these

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antibodies target, are required for uptake. RHAMM therefore may perform multiple

functions at the ce11 surface to mediate cell locomotion.

Cellular RHAMM levels have been shown to vary depending upon time in

culture. This prompted an examination of the relationship between cellular

RHAMM levels, and relative TR-HA uptake. Fipre 111.11. shows that TR-HA

uptake is at its highest in RHAMMAI-5 transfected cells at 12 hours, this correlates

nicely with previous results using flow cytometry which indicate that RHAMM

expression on the cell surface is highest upon 12 hours of plating (Chang et al. in

preparation). Further, among the three ce11 lines used in these experiments, TR-HA

uptake is shown to positively correlate with functional (Le. RHAMM which is

capable of binding HA) cellular RHAMM levels at al1 tirnepoints (Figure 111.11.).

RHAMM has been proposed to regulate gap junctional intercellular

communication through connexin43 (Nagy et al. 1995), and a potential role for gap

junctional transport in TR-HA trafficking was investigated using connexin43

transfected gliorna cells. At confluence it can be seen that connexin43 transfected

cells show a greater amount of intracellular TR-HA than their parental glioma cells

and individual connexin43 transfectants appear to target TR-HA to the tips of cell

processes, while their parental glioma cells do not (Figure 111.12.). These results are

consistent with a role for gap junctions in TR-HA targeting, but it is not possible to

determine if this is a specific effect resulting from functionally competent gap

junctions between cells or hernigap junctions (Ebihara et al. 1995) at the plasma

membrane, or one reflecting other factors which are upregulated upon connexin43

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transfection. Future experiments are needed to determine the role of connexin43 in

TR-HA uptake.

In a recent study HA treatment of rat embryonic cell Iines, under similar

conditions of confluence, plating time, and HA concentrations/times as those used in

this study, has been shown to rapidly activate tyrosine phosphorylation of cellular

proteins and MAP kinase in a time and dose dependent manner, and subsequently

stimulate cell growth (Serbulea et al. 1 999). This HA dependent activation of MAP

kinase was strongly suppressed by dominant negative ras expression (Serbulea et al.

1999). These results suggest that the ras-MAP kinase pathway is activated by HA

and may play an important role in HA dependent signaling (Serbulea et al. 1999).

Our lab has demonstrated that the HA binding domains of intracellular RHAMM are

required for interaction in MAP kinase signaling (Zhang et al. 1998). and these

results collectively suggest a role for intracellular HA in the MAP kinase cascade.

Conversely MEK inhibition with PD098059, which prevents the activation of MEK

by Raf in this cascade (Alessi et (11. 1995), appears to inhibit TR-HA internalization

(Figure II1.13.), and would suggest that HA internalization is somehow dependent

upon MAP kinase signaling. This effect may be due to multiple effects of the MEK

inhibitor, including hyperpol ymerizaton of microtubules (Harrison and Turley in

preparation) with consequent interference with vesicle rnediated transport of HA

dong microtubules (see Appendix 1 - Figure VI. 1 .). MEK inhibition also causes

decreased cell motility (Paiwand et al., in preparation), which may have far reaching

implications in decreasing levels of overall cellular activity and HA uptake.

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IV.2. Potential roles for intracellular HA targeting

IV.2.1. Potential role of HA in the nucleus

Precedent for regulatory roles of nuclear GAGs corne from studies showing

that transient nuclear heparin sulfate is invol ved in growth control and transcriptional

regulation (Fedarko and Conrad 1986; Fedarko et al. 1989; Ishihara and Conrad

1989; Busch et al. 1992). Exogenous addition of heparin to cells has been show11 to

arrest the ce11 cycle in G 1 (Fedarko et al. 1989). stimulatelinhibit DNA synthesis in

nomal/tumor cells (Furukawa and Bhavanandan l982), compete for binding with

histones (Fumkawa and Bhavanandan 1983; Scheerer et al. 1989). suppress the

expression and DNA binding actions of AP- 1 (Au et al. 1994) and accumulate at the

nuclear membrane (Fedarko and Conrad 1986). Though multiple GAGs including

HA, CS, HS and heparin have d l been reported to be associated with chromatin in

nuclei (Furukawa and Terayama 1977), heparin has so far received more attention for

possible direct regulatory roles.

We show here a novel import of exogenous HA into the nucleus, and its

distribution appears ordered (Figures 111.1.-111.2.). Other studies have reported the

presence of endogenous HA in the nucleus (Margolis et al. 1976; Fumkawa and

Terayama 1977; 1979; Castejon and Castejon 1987; Ripellino et al. 1988; Kan 1990;

Eggli and Graber 1995; Evanko and Wight Ni press). Within the nucleus HA has

been found associated with nucleoli (Kan 1990; Evanko and Wight in press),

heterochromatin (Eggli and Graber 1995), fine filaments within the nuclei (Evanko

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and Wight in press) and in nuclear clefts and surrounding condensed chromatin

during rnitosis (Evanko and Wight in press). GAGs have been found associated with

DNA in loops, many in aggregates or cycles resembling the pore complexes, which

suggests evidence for a structural role for GAGs in the nucleus (Engelhardt et al.

1982). As well, nuclear GAGs appear increased during liver regeneration (Funikawa

and Terayama 1977). The functional importance of these findings is not known.

The recent report of a cytoplasmic HA binding protein p68 which has been

CO-purified with the splicing factor SF2 (Deb and Datta 1996), as well as the

existance of other hyaladherins in the nucleus including chick ce11 cycle homologue

cdc37 (Gramrnatikakis et al. 19951, and RHAMM, al1 indicate the potential for a

multifaceted control of cell function by HA from within the nucleus.

Aside from its protein interaction, HA has the potential to exert influence in

the nucleus through basic biochemical and structural modalities. HA is a calcium

chelator and could potentially be involved in regulating nuclear calcium levels,

which control a variety of nuclear functions, including gene transcription, DNA

synthesis, DNA repair and nuclear envelope breakdown (Santella and Carafoli 1997).

The nucleus is a dynamic structure, with chromosome movements, and cycles of

condensation and decondensation (Larnond and Earnshaw 1998). The hydrating and

basic physicochemical properties of HA suggest that HA may be involved in

facilitating transport of small molecules within the nuclear interior just as it does in

the ECM.

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How or whether HA might interact with nuclear pores remains unclear. but a

growing body of research on nuclear import of glycoconjugates suggests that when

proteins are substituted with 25 GlcNAc residues (linked in p 1-4 fashion), they are

pennitted to be transported from the cytotosol to the nucleus (Duverger et ni. 1993;

1995; 1996). This form of nuclear transport has been shown to use the nuclear pore

in a novel fashion which does not compete for cofactors associated with traditional

NLS dependent protein import (Duverger et al. 1995). It is possible that this form

of transport could be responsible for mediating the novel nuclear translocation of HA

seen in this study.

The size of HA preseni in both the nucleus and cytoplasm remains open for

speculation. Recently identified GPI linked ce11 surface hyaluronidases (Chan et al.

1999), and potential hyaluronidases within the cell may act to cleave HA as it is

being transported into the cell. Localization of HA in the nucleus using a

biotinylated aggrecan probe (Eggli and Graber 1995; Evanko and Wight in press)

demonstrates that the HA within the nucleus must be at least HAIO. since aggrecan

HABP requires at least a decasaccharide of HA to recognize and bind HA (Hascall

and Hinegard 1974).

Surprisingly, preliminary evidence has identified the nucleus as the

predominant cellular locdization of the newly characterized hyaluronidase HYAL 1

in normal epithelium both in vitro and in vivo (Frost et al. in preparafion). HYALl

is a candidate tumor suppressor gene (Frost et al. 1997), as it is found at the site of a

common tumor suppressor locus (Killary et al. 19%; Buchhagen 1996). The

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putative localization of HYALl to the nucleus in normal epithelia has given insight

into possible mechanisms of how HYAL 1 might function as a tumor suppressor

gene. In addition, HYAL- 1 is unable to remove cell surface HA but may rather play

a functional role in the nucleus to remove nuclear GAGS that may regulate cellular

proliferation, differentiation, and or apoptosis (Frost et al. in preparation).

IV.3.2. Potential roles of HA uptake in transformation

HA production and metabolism are often altered during turnorigenesis

(Knudson and Knudson 1993) and a causal role for this modification has recently

been suggested by a study showing that HA accumulation around and within

colorectal tumor cells is prognostic of poor outcome (Ropponen et al. 1998).

Consistent with the notion that HA directly regulates cell behaviour, study of HA

binding proteins tenned hyaladherins has indicated that HA contributes to the control

of ceIl cycle and cell motility (Entwistle et al. 1996; Mohapatra et al. 1996; Wang et

al. 1998; Zhang et al. 1998). in spite of the importance of these observations, the

molecular mechanism by which HA directs these processes are not clear.

HA uptake was observed in five cell lines and levels of cellular

intemalization were shown to be highest in cells expressing the highest levels of

RHAMM, ras, CD44 and erk, as shown in both cells and invasive breast cancer cells

compared to their non transformed counterparts (Figure 111.4.). This trend

correlates with both increased motility and proliferation in these ce11 lines.

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HA oligosaccharides have been shown to significantly reduce prolifention

and migration in response to PDGF (Evanko et al. 19991, and to inhibit tumor

formation in subcutaneous rnelanoma cells (Zeng et al. 1998). Thcse effects may be

mediated ihrough blocking accumulation of HA in intracellular compartments

relevant to tumorigenesis.

The potential uses of covalent! y modified HA for selectivel y targeting and

accumulating in aggressive cancer cells are manifold in both diagnostics and

treatment. One recent study used HA linked to sodium butyrate to target HA to

breast cancer cells and found that cells could internalize both HA and the covalently

linked compound, which resulted in inhibition of cellular growth rates (Coradini et

al. 1999).

IV.3.3. Potential roles of intracellular HA in cell motility

The ability of HA microinjected into cells that do not aggressively take up

HA or target it into the cell processes, to stimulate ceil motility provides the first

direct evidence of an effect of intracellular HA on ce11 behaviour (Collis et al. 1998).

Results in this study funher suggest that the uptake and intracellular targeting of HA

to cell processes is controlled by ras and PKC signaling pathways. lOT1/2 cells

which are not transformed or treated with PKC do not take up HA aggressively, and

do not target HA to the tips of ce11 processes (Figure III.14.a.). These same cells

will not respond to exogenous administration of HA with increased motility - as ras

and RHAMM transformed cells do (Collis et al. 1998). However, when 10T112 cells

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are treated with phorbol ester. exogenous HA is targeted to the tips of ce11 processes

(Figure III.14.b.). concomitant with manifold increased motility (Collis et al. 1998).

The ability of HA to be directed into 10T1/2 ce11 processes after acute

phorbol ester treatments suggest an involvement of protein phosphorylation by

protein kinase C in this HA uptake. This protein serine/threonine kinase has

previously been implicated in cell motility (Liu et al. 1997) and its kinase activity

has also previously been linked to release of HA from the ce11 (Klewes and Prehm

1994; Thiebot et al. 1999), which may involve analogous mechanisms to uptake

noted here.

The dramatic alterations in TR-HA subcellular targeting upon phorbol ester

treatrnent (Figure 111.14) may be mediated through PKC and ras dependent

activation of microtubule-dependent particle motility (Alexandrova et al. 1993).

PMA treatment has been shown to target transfemn receptors to cellular

compartments which do not participate in normal routes of receptor traffic, or to

otherwise inaccessible compartments along new membrane traffic pathways

(Schonhorn et al. 1995). It would likewise be interesting to examine the effects of

PKC inhibitors upon this process.

Further evidence for intracellular HA upregulation upon onset of motility is

shown in wounded monolayers, where TR-HA is shown to accumulate intracellularly

at the wound edge (Figure III.15.A.B.). This effect is most dramatically enhanced

within 30 minutes post-wounding (Figure III.IS.C.), concomitant with increased

RHAMM expression during early wound responses (Savani et al. 1995b). Further,

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wounding is shown to promote subcellular targeting of HA to the tips of cell

processes concomitant with their increasing motility (Figure III.1S.A.).

Previous reports have demonstrated HA accumulation at wound sites, both in culture

and in vivo (Gerdin and Hallgren 1997; Chen and Abatangelo 1999). but the

possibility that HA'S therapeutic activity at such sites is being mediated

intracellularly is a novel one.

IV.4. Conclusions

Wr note here that the rapid uptake of fluorochrome-tagged HA into cells

appears to be independent of the traditional receptor-mediated endocytic pathway,

since uptake is acute, is incompletely blocked by CD44 antibodies, and results in the

accumulation within ce1 1 processes, the nucleus, and the pennuclear area. The

localization of HA in the nucleus requires an interaction with subcellular proteins,

since exposing cells to peptides that mimic HA binding motifs of RHAMM block

this targeting and results in accumulation of HA only within the cytoplasm (Figure

111.9.). HA binding motifs (Yang et al. 1994) are found in RHAMM, cdc37. and

p68, which are d l potential candidates involved in the uptake of HA. Our results

also suggest the targeting of HA to ce11 processes involves a mechanism distinct

from nuclear targeting.

Collectively, Our results suggest that both HNcell surface receptor and

intracellular HNprotein interactions may be involved in regdation of ce11 behaviour.

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A multifaceted role of intracel1uIa.r HA is suggested by its localization in the nucleus,

previous reports of HA binding to nuclear proteins (Furukawa and Terayama 1977;

1979) and the possibility that a hyaluronan binding protein may interact with RNA

splicing machinery (Deb and Datta 1996). Our results provide the first preliminary

evidence of the ability of exogenously added HA to accumulate within multiple

subcellular compartments, and to directly affect ce11 motility. Further analysis is

required to determine the precise role(s) of HA within the cell, to define the

mechanisms involved in this uptake and accumulation process and to determine the

signaling processes that intracellular HA might regulate. Clearly, study in this field

is only just beginning, and more work is necessary to elucidate the mechanisms

mediating HA traffic, and the role(s) of HA inside the cell.

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CHAPTER V

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CHAPTER VI

APPENDIX 1 - ADDITIONAL RESULTS SECTION

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VI.1. Results

VI.1.1. Colchicine treatment prevents nuclear uptake of TR-HA

To examine the potential role of the microtubules in subcellular trafficking of

HA. cells were pretreated with colchicine to disnipt their microtubule networks, then

exposed to TR-HA. Cells pretreated with colchicine display TR-HA staining

diffusely throughout the nucleus and cell processes, while nuclear targeting of TR-

HA is ablated and perinuclear accumulation is decreased (Figure V1.1.).

VI.1.2. Brefeldin A pretreatment blocks uptake of TR-HA by lOTll2 parental

cells.

In order to mess the potentid role of vesicle trafficking in TR-HA transport,

treatrnent with Brefeldin A were used. Pretreatment of ras transformed and parental

IOTU2 cells wiih Brefeldin A prior to addition of TR-HA caused a dramatic

decrease in the intracellular accumulation of TR-HA in al1 cellular compartments

(Figure VI.2.).

VI.1.3. HA alters the ability of DNaseI to interact with DNA

DNase digestion of genomic DNA is enhanced in the presence of HA as

evidenced by increased smear length (corresponding to fragmented DNA) across an

agarose gel (Figure VI.3.). Another glycosaminoglycan, chondroitin sulphate has no

effect on DNA degradation, indicating that the process is HA specific (Figure VI.3.).

This phenornenon would appear to suggest that HA may promote interactions

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between protein and DNA on a wider basis, and suggests a fundamental role for HA

in the nucleus.

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Figure VI.1. RHAMMA 1-5 transfected cells treated with 1OmM colchicine for 30

minutes prior to addition of TR-HA (100pg/ml) and funher incubation for (a) IO

minutes or (b) 30 minutes show no TR-HA trafficking to the nucleus, in contrast to

non-colchicine treated cells at (c) 10 minutes and (d) 30 minutes TR-HA. Bars =

1 Opm.

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Figure VI.2. (a,b) Parental and (c,d) ras-transfonned fibroblasts pretreated with

(b+d) 50pg/rnl Brefeldin A for 30 minutes prior to TR-HA treatment (lOOClg/rnl; 10

minutes) show greatly decreased levels of TR-HA uptake compared to cells not

pretreated (a+c). Bar = 1 Opm.

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Fipre V1.3. Lanes 1-8 are loaded with genomic DNA degraded by increasing

concentrations (0.0 - 7.8 x 104 uniis) of DNaseI. Prebinding of DNA with 10pg

(lanes 9- 16) or 5Opg (lanes 17-24) HA results in increased DNA degradation by

DNaseI (as visuaiized by increased smear length). 10pg chondroitin sulfate (lanes

25-28) had no effect on DNA degradation by DNaseI. Samples loaded on a 1%

agarose gel.

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VI.2. Discussion

VI.2.1. Cytosolic factors in the uptake process

Cells which have had their microtubule networks disrupted by colchicine

have dramatically reduced transport of TR-HA to the nuclear and perinuclear

compartments, while entry of TR-HA into the cytoplasm and ce11 processes appears

unaffected (Figure VI.1.). This predicts a form of microtubule dependent transport

of HA across the cytoplasm, which is involved in targeting HA to sites relevant for

nuclear entry. Microtubules have been previously shown to direct a multistep

process of caveolae cycling between the Golgi and plasma membrane, and this

transport is intemipted by microtubule disruption using nocodazole (Conrad et al.

1995). Therefore we propose that at least part of the TR-HA transport could be

O C C U ~ ~ ~ via this rnicrotubule mediated transport of caveolae.

In previous studies, colchicine has been demonstrated to decrease levels of

cellular HA, potentially impinging upon HA synthesis (Brecht et al. 1986). Perhaps,

without an intact microtubule network, levels of HA accumulate at cellular and

intracellular sites which act to downregulate HA synthesis. Interestingly, addition of

HA has recentiy been shown to cause an increase in intracellular concentrations of

tubulin in human cells (Greco et al. 1 W8), and these results have implications for the

role of HA in ce11 division.

Vesicular transport within the ceIl is facilitated by recruitment of cytosolic

coat proteins that promote the budding of transport vesicles (Kreis and Pepperkok

1994). Brefeldin A inhibits recruitment of these coat proteins and formation of

transport vesicles. Brefeldin A has been shown to interfere with vesicular transport

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causing disassembly and collapse of Golgi components back to the ER (Hidalgo et

al. 1992). Disruption of vesicular transport with Brefeldin A causes a decrease in

overall intracellular accumulation of TR-HA (Figure VI.2.), implicating vesicular

mediated transport at both the plasma membrane, and in cytoplasmic trafficking of

HA. These effects are also consistent with a microtubule mediated vesicular mode

of TR-HA transport (perhaps associated with caveolae), to the perinuclear/nuclear

compartments (see Figure VI.1.).

VI.2.2. Potential influence of HA on DNA - protein interactions

HA has been found associated with the chromatin fraction of the nucleus

(Furukawa and Terayama 1977; 1979; Eggli and Graber 1995), and previous work in

Our lab has demonstrated that HA is capable of binding to genomic DNA (Zhao et al.

in preparation). We show that HA is able to increase the effectiveness of DNaseI

interactions with DNA, resulting in increased degradation of DNA (Figure VI.3.).

At this time it is not possible to speculate whether this specific ability of HA to alter

protein-DNA interactions is efficacious within ce11 nuclei, but it does present us with

an interesting mode1 for HA activity. Likewise, in the cytoplasm, it is possible that

HA influences protein-protein interactions. The HA binding domains of RHAMM

are also required for RHAMM-erk binding to occur (Zhang et al. 1998). It is

possible that HA cm compete with erk for these binding sites, and thereby influence

downstream events. Peptides which mimic HA'S ability to bind these domain have

been developed and are being used to investigate this possibility (Ziebell et al. in

preparation) .

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Figure V1.4. Model of potential uptake mechanisms and intracellular functions

of HA (see next page): HA uptake is potentially mediated by multiple processes:

( 1 ) Clathnn mediated HA uptake and targeting to lysosomes involves CD44

(Alston-Smith et al. 1992). (2) Caveolae rnediated pinocytic HA uptake rnay involve

RHAMM, CD44 and p68 proteins. This form of transport utilizes microtubules to

traffic vesicles to and from the golgi/ER pennuclear compartment. (3) The HA

synthase complex may at times direct HA cytoplasmically. (4) HA may interact with

the phospholipids of the cell membrane to form pores or channels (Pasquali-

Ronchetti et al. 1997), and thus enter the cell cytoplasm in a non energy dependent

fashion. Once internalized HA occurs notably at tips of ce11 processes upon onset of

motility as produced by ceIl transformation and PKC treatments. IntracelIular HA of

larger than HA30 also accumulates in ce11 nuclei. The presence of cytoplasmic and

intranuclear hyaladherins provide additionai functional targets for iniracellular HA.

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1. Degradative . Endocytic

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CHAPTER VI1

APPENDIX II. - HYALURONAN DETECTION

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VII.1. Existing probes for hyaluronan

The andytical techniques for HA detection have been continuously refined.

For many yean it was necessary to purify the polysaccharide so that its sugar

constituents, glucuronic acid and glucosamine could be measured by colour

reactions. A ciassical technique was the carbazole method for uronic acids. These

analyses required at least 100pg of HA. With the discovery of specific rnicrobial

enzymes that degrade HA into unsaturated disaccharides, it became possible to

design techniques in which a few micrograms of poiymer were sufficient. The third

generation came in 1980. These make use of proteins with specific affinity for HA

and can be used as antibodies in ELISA4 ke assays to detect as little as 1 ng of

polysaccharide.

More recently a technique for the histolocalization of HA (Ripellino et al.

1985) that utilizes these highly specific HA binding peptides has become widely

used. However, even this HABP procedure may senously underestimate the HA

content of tissues fixed in a routine manner (Lin et al. 1997). Other GAGS are fixed

as a result of their covalent binding to profeoglycan core proteins, however the free

form of HA apparently diffuses slowly into the fixation solution during the process.

Although CPC improves the preservation of HA in such samples, total HA cannot be

accounted for (Lin et al. 1997).

VII.2. The Texas Red - hyaluronan conjugate

Four groups on the HA molecule are available for chernical modification: the

carboxylate, the acetarnido, the reducing end-group and the hydroxy groups.

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Modification of HA with ethyl (N,N-dimethy1aminopropyl)carbodiimide (EDCI) has

been done since 1986 (Kuo et al. 1990). The productions of glucuronamides

requires the activation of the carboxylic group which can be accomplished using a

water soluble carbodiimide such as EDCI as the condensing agent.

Advantages include 1 ) preparation of HA derivatives under mild, aqueous

conditions which preserves the structural integrity and molecular size range of HA,

also 2) homogenous modified HA materials can be easily obtained by removal of al1

side products and unreacted reagents dunng dialsysis and 3) al1 new HA derivatives

based on this bishydrazide technology are chemicall y well-characterized (Prestwich

et al. 1998).

TR-HA can be prepared directly from HA and Texas Red sulphoylhydrazide

(TR-HA used in this study has been analyzed to have 1 in 15 disaccharides

successfully labeled withTR - Figure VII.1.). This gives maximal sensitivity while

preserving the minimum decasaccharide recognition unit of HA for HABDs. These

probes offer advantages over reducing end-modified HA derivatives (Raja et al.

1988; Yannarielio-Brown et al. 1996), in chat multiple probe moieties can be

included per HA rnolecule, and the probes are positioned in the intemal milieu of

HA.

Future directions in the chernical modification of HA include development of

covalent attachments of therapeutic dmgs to HA (Band et al. 1998). Attachment of

HA to proteins, short peptides, mti-cancer agents, anti inflammatory or anti infective

drugs or cell adhesion and growth pmmoting ligands provides the potential for drug

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delivery through exploitation of HA recognition and receptor-rnediated endocytosis

for specific uptake by and delivery to target cells (Vercruysse and Prestwich 1998).

a i+r.r6 I l t r l r r i

C W 1 i m.. L...,..,.

e..&. ?"*"i

Y, II. Y.. PID.".bU -*Yi -

Figure VII.1. HPLC analysis of TR-HA conjugate. Analysis of success of

conjugation of TR-HA: the (a) refractive and (b) absorptive (at 590nm) indices

overlap at 19 minutes elution time in a single peak indicating the sample is pure.

(c) Molecular weight of the HA is shown to be approximately lOOkDa through

cornparison to other HAs of known molecular weights. To measure success of

conjugation (d) comparative absorbance of the TR-HA on a TR standard curve

shows that approximately 1 in 15 HA dissaccharides has been successfully

labeled.