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Tiêu đề Amyloid oligomers: diffuse oligomer-based transmission of yeast prions
Tác giả Hideki Taguchi, Shigeko Kawai-Noma
Trường học University of Tokyo
Chuyên ngành Medical Genome Sciences
Thể loại Minireview
Năm xuất bản 2010
Thành phố Kashiwa
Định dạng
Số trang 10
Dung lượng 361,45 KB

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Yeast prion [PSI+] Although several dozen yeast proteins are known to behave as prions in vivo for recent advances, see refs 9-12, the prion state of the Sup35 protein – the [PSI+] deter

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Amyloid oligomers: diffuse oligomer-based transmission of yeast prions

Hideki Taguchi and Shigeko Kawai-Noma

Department of Medical Genome Sciences, Graduate School of Frontier Sciences, University of Tokyo, Kashiwanoha, Kashiwa, Chiba, Japan

Introduction

Scrapie in sheep, bovine spongiform encephalopathy

(also called ‘mad cow’ disease) in cattle and

Creutz-feldt–Jakob disease in humans are transmissible

spong-iform encephalopathies, which are also called prion

diseases A prion is a proteinaceous infectious particle

that lacks nucleic acids, which means that it is an

infectious protein [1] In the prion, the altered

con-formers of a protein autocatalytically convert the

nor-mal structure to the altered form, which is an ordered

aggregate called amyloid Although this prion concept

was developed for the mammalian neurodegenerative

diseases in which the PrP protein participates, the

con-cept has been extended to several non-Mendelian

genetic elements in budding yeast, such as [PSI+] and

[URE3] in Saccharomyces cerevisiae [2] As yeast is

quite a tractable model eukaryote, yeast prions can provide many important insights, which are usually difficult to achieve using mammalian prions, into prion biology [3–8] In particular, the molecular mechanisms

by which the prion proteins are propagated and trans-mitted have been unraveled in the yeast prion model This review provides an overview on the transmissible entities of prion proteins in yeast prion [PSI+]

Yeast prion [PSI+]

Although several dozen yeast proteins are known to behave as prions in vivo (for recent advances, see refs 9-12), the prion state of the Sup35 protein – the [PSI+] determinant – is the best-characterized prion

Keywords

amyloid; fluorescence correlation

spectroscopy; prion; Sup35; yeast prion

Correspondence

H Taguchi, Department of Medical Genome

Sciences, Graduate School of Frontier

Sciences, University of Tokyo, FSB401,

5-1-5 Kashiwanoha, Kashiwa, Chiba, 277-8562,

Japan

Fax: +81 4 7136 3644

Tel: +81 4 7136 3644

E-mail: taguchi@k.u-tokyo.ac.jp

(Received 4 September 2009, revised 24

November 2009, accepted 25 November

2010)

doi:10.1111/j.1742-4658.2010.07569.x

Prions are infectious proteins, in which self-propagating amyloid conforma-tions of proteins are transmitted The budding yeast Saccharomyces cerevi-siae, one of the best-studied model eukaryotes, also has prions, and thus provides a tractable model system with which to understand the mechanisms

of prion phenomena The yeast prions are protein-based heritable elements, such as [PSI+], in which aggregates of prion proteins are transmitted to daughter cells in a non-Mendelian manner Although the genetic approaches preceded the yeast prion studies, recent investigations of the dynamic aspects of the prion proteins have unraveled the molecular mechanisms by which prions are propagated and transmitted In particular, several lines of evidence have revealed that the oligomeric species of prion proteins dispersed in the cytoplasm are critical for the transmission This review summarizes the topics on the transmissible entities of yeast prions, focusing mainly on the Sup35 protein in [PSI+]

Abbreviations

FAF, fluorescence autocorrelation function; FCS, fluorescence correlation spectroscopy; FRAP, fluorescence recovery after photobleaching; GFP, green fluorescent protein; GuHCl, guanidine hydrochloride.

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Sup35 is an essential protein, which functions as a

translation termination factor in cooperation with its

partner, Sup45 [13] The N-terminal portion of Sup35

is a glutamine⁄ asparagine (Q ⁄ N)-rich domain, which

has a high propensity to form amyloid fibrils in vitro

[14–18] (Fig 1) The C-terminal domain is sufficient to

function as the termination factor (eRF3) and interacts

with Sup45 (eRF1) [13] The [PSI+] phenotype is a

nonsense suppression caused by the amyloid-like

aggregates of Sup35 [13] The propagation of [PSI+] is

strictly dependent on the presence of an appropriate

amount of Hsp104 [19] Impairment of Hsp104

func-tion, by either the deletion of Hsp104 [19] or the

addi-tion of millimolar concentrations of guanidine

hydrochloride (GuHCl), cures [PSI+] [20]

One of the central issues in prion biology is to

iden-tify the entity of the [PSI+] determinant Several

approaches, as described below, have been applied

over the past decade

Biochemical approaches to investigate

yeast prion aggregates

The proposal that non-Mendelian genetic elements

(such as [PSI+]) in yeast are prions has opened the

door to identify the molecular entity of prions [2] The

aggregates of Sup35 in the cells can be detected by

the centrifugation of yeast lysates [21,22] However,

the centrifugation assay cannot address the detailed

characteristics of the Sup35 aggregates, such as the

structure and size of prion aggregates in vivo

Kryndushkin et al developed an ingenious method

to characterize the prion aggregates in cells They

found that treatment of the [PSI+] lysate with 2% SDS disassembles the Sup35 prion aggregates into smaller, SDS-resistant particles (called polymers in their report), allowing analysis of their sizes on an aga-rose gel containing 0.1% SDS [23] This method is referred to as semidenaturing detergent–agarose gel electrophoresis [23] Using this method, they compared [PSI+] and [psi)] lysates and found that Sup35 was almost always in the oligomeric form in the [PSI+] lysate, while it was monomeric in the [psi)] lysate They concluded that the SDS-resistant prion oligomers were heterogeneous in size, ranging from 700 to

4000 kDa, which should correspond to 8-50 Sup35 monomers [23]

The possibility of other interacting proteins being present in the Sup35 oligomers was tested by affinity isolation of Sup35–His6 in [PSI+] cells [24] The affin-ity isolation revealed that the SDS-resistant Sup35 oligomers are associated with Ssa1⁄ 2 proteins in a molar ratio of 0.5 Ssa1⁄ 2 per Sup35, and with other minor components, including Hsp104, Ssb1⁄ 2, Sis1, Sse1, Ydj1 and Sla2 [24] When the affinity-purified Sup35 oligomers were negatively stained and visualized

by electron microscopy, the oligomers resembled short barrels and bundles, which seemed to be composed of barrels, rather than long fibrils [24]

Genetic approaches to investigate the transmissible entity of yeast prions

As mentioned earlier, the addition of GuHCl leads to the elimination of [PSI+] through the inhibition of Hsp104 activity [20,25–29] The kinetics of prion elimi-nation exhibited a significant lag, corresponding to around four to five generations, before the gradual emergence of [psi)] [25] Based on the kinetics, Tuite and co-workers proposed that GuHCl blocks a critical step in the replication of the prion conformers [25,30] Assuming that the prion seeds are randomly segre-gated during cell division, they calculated that the number of prion seeds, which have been named

‘propagons’, in [PSI+] cells was approximately 60 [30] Sophisticated yeast genetics revealed that [PSI+] cells contain propagons to transmit and maintain the prion phenotype

Green fluorescent protein-fused Sup35 aggregates as an indicator of [PSI+] cells

The expression of Sup35 fused with green fluorescent protein (GFP) in [PSI+] cells gives rise to the forma-tion of visible spherical fluorescent aggregates, called

Fig 1 Electron micrograph of amyloid fibrils formed by yeast prion

Sup35 Negatively stained amyloid fibrils formed by the

recombi-nant NM fragment of Sup35 [18] Bar indicates 50 nm.

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foci, in the cytosol [22] (Fig 2A), although the size

and the number of foci vary, depending on the

expres-sion level and the yeast strains used Besides the

spher-ical foci, ring⁄ rod-shaped aggregates were also

observed under some conditions, such as in [PSI+]

cells treated with GuHCl [31,32] (Fig 2B) By contrast,

Sup35–GFP does not form such visible foci, and is

dif-fusely dispersed in the [psi)] cells [22] (Fig 2C) Most

of the Sup35 in the [PSI+] cell lysate exists as

high-molecular-weight pellets, and is isolated by high-speed

centrifugation [21,22] In vitro, Sup35 fragments

containing the N domain form b-sheet-rich amyloid

aggregates [14–18] Therefore, the spherical foci are

considered to be an indicator of [PSI+] because no

such foci are seen in [psi)] cells

Regarding the relationship between the visible

Sup35–GFP foci and [PSI+], Zhou et al [31]

observed the formation of Sup35 aggregates in vivo

during the de novo induction of [PSI+] by the

overex-pression of Sup35 in [psi)][PIN+] cells, where [PIN+]

is a yeast prion phenotype that requires the induction

of [PSI+] [33] Based on their detailed examination of

the appearance of the visible aggregates in mother

and daughter cells, as well as analyses of the timing

of aggregate formation, they suggested that most of

the heritable [PSI+] seeds are too small to be

visual-ized by conventional fluorescence microscopy [31]

Song et al [34] also reported that fluorescent foci

do not directly represent [PSI+], based on the

care-ful observation of visible foci in [GPSI+] cells, in

which GFP was inserted between the N and M

domains of Sup35 in the chromosomally encoded

SUP35gene

Studying the dynamics of Sup35–GFP aggregates in living cells

Prion phenomena are intrinsically dynamic processes, because prion aggregates propagate, remodel and transmit during the protein-based inheritance in yeast prions [3,8] Simple static observations of Sup35–GFP aggregates by conventional fluorescence microscopy are insufficient to investigate the dynamic aspects of the prions in the cells Recent advances using several techniques to investigate the dynamics of protein mole-cules in living cells have provided novel insights into the molecular mechanism by which the Sup35 prion aggregates are propagated and transmitted in [PSI+] cells

a) Single-cell imaging system to monitor the fate

of the Sup35–GFP foci Several genetic analyses, combined with fluorescent microscopic observations of visible Sup35–GFP foci, have suggested that the foci do not directly represent [PSI+] However, such an ensemble method does not provide direct evidence for the significance of the visi-ble foci in the transmission of [PSI+] To gain insight into the dynamics of Sup35–GFP foci in [PSI+] cells,

an on-chip single-cell cultivation system was developed

to investigate directly the dynamic properties of prion aggregates [35,36] Specifically, the fate of the visible Sup35–GFP foci in single living [PSI+] cells was directly monitored in a time-lapse manner The on-chip cultivation system, in which the medium can

be easily exchanged during cultivation, enabled us to

Fig 2 Sup35–GFP aggregates in [PSI+] cells (A) Expression of a Sup35–GFP fusion protein in a [PSI+] cell leads to the appearance of visible spherical aggregates (foci) Phase contrast (top) and fluorescent (bottom) images are shown (B) Rod-shaped or ring-shaped visible aggre-gates are formed under the conditions where Sup35NM–GFP was overexpressed in a [PSI + ] cell that was treated with 3  5 m M guanidine hydrochloride (GuHCl) (C) Expression of Sup35NM–GFP in a prion-free [psi)] cell results in diffuse fluorescence in the cytosol Bars indicate

2 lm.

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continuously observe individual growing cells for long

periods of time The expression of Sup35–GFP was

transiently induced to lead to the formation of visible

foci in the cytoplasm of [PSI+] cells [35] After

stop-ping the further induction of Sup35–GFP, by

exchang-ing the medium with one that lacks an inducer, the

fate of the fluorescent foci was monitored in real time

Individual live-cell imaging showed that the diameter

of the foci gradually decreased, and the foci eventually

disappeared [36] The disappearance of the foci was

not caused by GFP photobleaching or degradation

[36] The disappearance of visible foci of Sup35–GFP

was also reported by analyses of a microcolony assay

system, in which preformed Sup35–GFP foci became

undetectable as the cells grew [37]

The punctate foci reappeared when Sup35–GFP

was re-induced, indicating that the seeds of the foci

were not lost in the cells after the foci disappeared

The live-cell imaging showed the appearance of the

foci in the daughter cell at almost same time as when

the foci re-appeared in the mother cell, indicating

that the seeds are transmitted from the mother cell

to the daughter cell In addition, several lines of

evidence, including a nonsense suppression assay at a

single-cell level, showed that the [PSI+] phenotype

was maintained in the cells after the foci dispersed

[36] Taken together, the single-cell imaging of

Sup35–GFP foci clearly revealed that the foci

dynam-ically dispersed into a state that functions as the

seeds of the foci and causes the nonsense

suppres-sion, which is sufficient to maintain the [PSI+]

phenotype

b) Fluorescence correlation spectroscopy

The single-cell imaging described in the previous

sec-tion revealed that, after dispersion of the foci, the

[PSI+] cells have an entity that behaves as prions

The next question is, what is the prion entity left in

the cytoplasm after the foci have dispersed?

Conven-tional fluorescence microscopic observation can barely

distinguish the difference between [PSI+] cells

with-out the foci and [psi)] cells in their appearance, as

both cells have diffuse GFP fluorescence in the

cytoplasm To elucidate the physical properties of

Sup35–GFP in living [PSI+] cells without the foci,

fluorescence correlation spectroscopy (FCS) has been

applied [36]

FCS is a technique used to analyze the diffusion

properties of fluorescent molecules, by calculating the

fluorescence autocorrelation function (FAF) in a

microscopic detection volume at the femtoliter level

[38,39] FCS allows the determination of diffusion

constants, which are directly correlated with the size of the molecules, of fluctuating fluorescent molecules under equilibrium conditions The dynamic range of FCS is very wide: FCS can measure commonly accessed diffusion dynamics on a timescale from

 1 ls to  1 s Because FCS is usually combined with confocal laser-scanning microscopy, we can define the detection volume at any position of interest inside

a living cell, in a non-invasive manner As the dynam-ics of prions are basically dependent on the conversion from monomers to aggregates, and vice versa, FCS is ideally suited to estimate the size of Sup35–GFP in living yeast cells

After confirming that FCS was applicable to living yeast cells, fluorescence fluctuations of Sup35–GFP in [psi)] and [PSI+] cells, with or without the foci, were measured using FCS [36] The FAFs of Sup35–GFP

in [psi)] cells were almost the same as those in cells expressing the GFP monomer alone, indicating that [psi)] cells contain mostly monomers of Sup35–GFP

By contrast, the FAF profiles in [PSI+] cells, irre-spective of the presence of foci, were shifted to the right, compared with those in [psi)] cells, indicating that the Sup35–GFP species in [PSI+] cells were much slower, and thus larger, than those in [psi)] cells These results indicate that the larger species, referred to here as diffuse oligomers, are dispersed in the cytoplasm of [PSI+] cells, regardless of the pres-ence of foci [36]

The combination of FCS with the on-chip single cul-tivation system (a time-lapse FCS system) allows mea-surements of the size of Sup35–GFP in the daughter cells immediately after the transmission from the mother [PSI+] cells [36] Autocorrelation functions of both the mother and daughter cells were measured as the [PSI+] cell with the foci was budding Strikingly, the autocorrelation function of Sup35–GFP in the daughter cell in an early budding step was almost the same as that in the mother cell, indicating that the dif-fuse oligomers are transmissible to daughter cells [36] These time-lapse FCS experiments, combined with the retention of the seeds of the foci in the daughter cells, demonstrated that the oligomeric species dispersed in the mother cells are directly transmitted to their daughter cells [36]

The single mother–daughter pair analysis using FCS was extended to investigate the effect of Hsp104 on the transmission of Sup35–GFP [32] An FCS analysis

of GuHCl-treated [PSI+] cells revealed that Sup35– GFP diffusion in the daughter cells was faster; that is, the Sup35–GFP particle was smaller than that in the mother cells under the Hsp104-inactivated conditions [32] (see below for details)

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c) Fluorescence recovery after photobleaching

As an alternative approach to analyze the protein

dynamics in living cells, the diffusion of a fluorescent

protein can be measured using a photobleaching

tech-nique known as fluorescence recovery after

photoble-aching (FRAP) [38] In this technique, fluorescent

molecules in a small region of the cell are irreversibly

photobleached by transient exposure to a laser beam,

and the subsequent recovery of fluorescence in the

photobleached region is recorded [38] The fluorescence

intensity recovers when the fluorescent molecules

dif-fuse, as the bleached molecules diffuse away and the

unbleached molecules diffuse into the irradiated

region The kinetics of the recovery provides

informa-tion about the diffusion property of the molecules:

fas-ter recovery means fasfas-ter motion, indicating the

greater diffusion constant of the molecules As the

exposure to the laser beam for the bleaching is only

transient, this method is usually not harmful to living

cells The technique usually involves the production of

a specific protein of interest fused to GFP or other

fluorescent proteins, and has been applied to the

Sup35–GFP fusion proteins in living yeast cells

[32,34,37,40–42]

A modified [PSI+] strain, in which a functional

Sup35–GFP fusion protein (referred to as NGMC)

was created by introducing GFP between the

N-termi-nal and middle domains of endogenous Sup35, was

used for the FRAP analysis to measure the diffusion

of Sup35–GFP proteins [34] The FRAP analysis

showed that the fluorescence recovery was slower in

[PSI+] cells than in [psi)] cells, indicating that the

NGMC proteins were in an aggregated form in [PSI+]

cells [34] In a subsequent study, FRAP was used to

monitor the NGMC states in GuHCl-treated [PSI+]

cells [40] The cytoplasm in the cells showed a slower

rate of recovery after 1 hour of incubation in GuHCl,

but the rate of FRAP increased after 5 h of incubation

in the GuHCl-containing medium, and became

identi-cal to the rate observed in [psi)] cells [40] In an

inde-pendent study, FRAP was also used to measure the

physical state of Sup35–GFP in the Hsp104-inactivated

[PSI+] cells, by either the Hsp104 mutant or the

Gu-HCl treatment [37] The measurements indicated that

Sup35–GFP became largely immobile, with no

recov-ery of fluorescence in the Hsp104-inactivated cells [37]

This immobility was interpreted as a cause of the

seg-regation bias of Sup35 aggregates in the

Hsp104-inacti-vated cells, eventually leading to the loss of [PSI+]

[37]

Transmission of Sup35 from mother cells to

daugh-ter cells is critical in the prion phenomena Neither

conventional FRAP nor FCS can be used to investi-gate the flux of Sup35 between mother cells and daughter cells; however, a technique based on FRAP has been developed to investigate the flux [32] In the conventional FRAP technique, fluorescent proteins in

a small region of the cell are photobleached In the modified FRAP technique, the GFP fluorescence in the whole daughter cell is photobleached to assess the flux rate from the mother cell to the daughter cell When the modified FRAP, called MD-FRAP (mother

to daughter), was conducted with the [PSI+] cells, the flux of Sup35NM–GFP in the [psi)] cells was faster than that in the [PSI+] cells, reflecting the existence of diffuse oligomers of Sup35–GFP in the [PSI+] cells The MD-FRAP in the GuHCl-treated [PSI+] cells yielded two distinct distributions of the flux rates About half of the cells transmitted the Sup35NM– GFP with a flux rate that was almost identical to that

in the [psi)] cells, suggesting that the cells were already cured By contrast, either no, or extremely slow, flux was observed in the other half of the cells, reflecting the severe impairment of the mother–daughter trans-mission in the cells [32]

Dynamic properties of the diffuse oligomers in the [PSI+] cells

After the extension of the prion concept into yeast non-Mendelian genetic elements, a variety of tech-niques, as described above, have unraveled the molecu-lar entity of yeast prions, such as the [PSI+] determinant, over the past decade Together, the data from centrifugation assays, biochemical isolation, yeast genetics, GFP fusion methods and several single-cell approaches have revealed that the transmissible entities

of [PSI+] are the oligomeric states of Sup35 within the cytoplasm Importantly, the nature of the diffuse oligo-mers is not static, but highly dynamic Although the role of protein dynamics in prion propagation was well summarized in a recent review [8], we will further dis-cuss the details of the diffuse oligomers in the prion transmission, based mainly on our recent findings

a) Sup35–GFP foci are in equilibrium with diffuse oligomers in [PSI+] cells

Analyses using the on-chip single-cell imaging and FCS revealed the dynamics of visible foci derived from Sup35–GFP The visible fluorescent foci, which are one of the indicators of the [PSI+] phenotype, are dis-persed throughout the cytoplasm as diffuse oligomers, which are sufficient to maintain the [PSI+] phenotype [36] In addition, the diffuse oligomers are transmitted

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to their daughter cells, where the foci can reappear

[32,36] Taken together, the foci are not dead-end

aggregates, but are highly dynamic species that are in

equilibrium with the diffuse oligomers

b) What is the molecular structure of the diffuse

oligomers?

What is the molecular structure of the diffuse oligomers?

The in vitro properties of recombinant Sup35 proteins,

which form amyloid fibrils with cross b-sheet structures,

led us to hypothesize that the amyloid structures of

Sup35 are critical for the propagation and transmission

of [PSI+] in vivo However, there is no direct connection

that links the in vitro amyloid fibrils with the in vivo

transmissible entities of the prions Nevertheless, several

lines of evidence support the proposal that [PSI+] cells

contain amyloid structures of Sup35 First, in vivo,

Sup35 aggregates in [PSI+] cells are stained by an

amy-loid-staining dye, thioflavin S, indicating the presence of

cross b-sheet structures in [PSI+] cells [43] Second,

amyloid fibrils prepared from recombinant Sup35 in vitro

can efficiently convert cells from [psi)] to [PSI+] after

incorporation of the in vitro fibrils into the [psi)] cells

[44,45], indirectly showing that amyloid fibrils are

prop-agated in the [PSI+]-converted cells Third, an electron

micrographic analysis of Sup35 oligomers isolated from

[PSI+] cells revealed barrels 20 nm wide and larger

structures (bundles) [24] Although the appearance of

these structures does not resemble the typical amyloid

fibrils formed in vitro, it has been pointed out that these

structures look similar to the prion oligomers made of

recombinant Sup35, when prepared in the presence of

Hsp104 plus ATP [46] Fourth, a simulation of the FCS

data on Sup35–GFP oligomers in the [PSI+] lysate,

based on a semidenaturing detergent–agarose gel

electrophoresis analysis, clearly showed that the diffuse

oligomers are not spherical, but adopt a rod shape

(C G Pack, S Kawai-Noma, et al manuscript in

prep-aration), suggesting that the diffuse oligomers are in

amyloid-like structures Finally, electron micrographic

observations of GuHCl-treated [PSI+] cells, in which

large rod-like or ring-like aggregates of Sup35–GFP

were visible, showed that the visible rod-shaped

aggre-gates are composed of bundled fibrils (S Kawai-Noma,

A Hirata et al., manuscript in preparation) The

diame-ters of the fibrils in cells closely resemble those of

amyloid fibrils formed in vitro The bundled structure is

considered to be a consequence of the impaired Hsp104

function, which results in the formation of longer fibrils

that bundle together to form the rod-shaped structure

Taken together, the diffuse oligomers in [PSI+] cells are

most likely to be fragmented amyloid fibrils

c) Balance between growth and division of the diffuse oligomers

Prion propagation involves two distinct steps: a growth phase in which the existing amyloid particles elongate

in a self-catalyzed manner; and a division (or fragmen-tation) phase in which the amyloid particles are divided for multiplication [3] Stable maintenance of the prion phenomena relies on a delicate balance between the growth and division phases, resulting in the dynamic properties of prion particles (Fig 3) The details of the growth phase in vivo are poorly understood By contrast, the division phase has been extensively investigated because trans-acting Hsp104 and other chaperones play a critical role in this pro-cess Hsp104 is a member of the AAA+ superfamily

of ATPases (ATPases associated with various cellular activities), which is not required under normal growth conditions but is critical for surviving extreme stress, such as temperatures of 50 C [47] Hsp104, with the aid of the Hsp70⁄ 40 system, breaks protein aggregates

in an ATP-dependent manner [47] Perturbation of cel-lular Hsp104 levels dramatically affects the mainte-nance of [PSI+]: overexpression, inactivation and deletion of Hsp104 cure [PSI+] [19] Although the molecular basis by which Hsp104 overexpression cures [PSI+] remains to be solved, accumulating evidence

Fig 3 Dynamics of the yeast prion Sup35 in the cell Prion propa-gation involves two distinct steps In the growth phase, pre-existing amyloid particles elongate in a self-catalyzed manner After transla-tion at the ribosomes, Sup35 monomers (green circles) are incorpo-rated into the pre-existing amyloid fibrils (red arrowheads) In the division (or fragmentation) phase, the amyloid particles are divided for multiplication Stable maintenance of the prion phenomena relies on the delicate balance between the growth and division phases, resulting in the dynamic properties of prion particles.

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has revealed a mechanism by which the inactivation of

Hsp104 cures [PSI+]

First, microcolony observations and FRAP revealed

that the Sup35 aggregates became immobile as a result

of their increased size upon Hsp104 inactivation [37]

Second, the rod-shaped Sup35–GFP aggregates were

accumulated in GuHCl-treated [PSI+] cells [31,32],

probably because of an insufficient fragmentation of

Sup35 amyloid fibrils, which was caused by impaired

Hsp104 function Third, a single mother–daughter pair

analysis using FCS in GuHCl-treated [PSI+] cells

showed that Sup35–GFP diffusion in the daughter cells

was faster, that is, the Sup35–GFP was smaller, than

that in the mother [PSI+] cells, and it eventually

reached the diffusion profiles found in [psi)] cells [32]

Finally, MD-FRAP revealed that the flux of the

dif-fuse oligomers in the GuHCl-treated [PSI+] cells was

completely inhibited [32]

Taken together, these studies indicate that the

Hsp104 inactivation causes the severe transmission bias

between mother cells and daughter cells [32,37] In

other words, inactivation of Hsp104 alters the

dynam-ics of the diffuse oligomers of Sup35 by disrupting the

delicate balance to maintain [PSI+], eventually curing

[PSI+] So far, the mechanism by which Sup35

proteins are transmitted to daughter cells is unclear

Is there an energy-dependent transmission system, such

as the actin cytoskeleton? Alternatively, simple

diffu-sion might suffice for the transmisdiffu-sion to the daughter

cells We can assume a diffusion barrier at the bud

neck, in which the transmission of large Sup35

oligo-mers or long amyloid fibrils is more or less restricted

by an unknown mechanism

d) Size-dependent transmission of the diffuse

amyloid oligomers

Recent data on the protein dynamics under the

Hsp104-perturbed conditions prompted us to propose

that the propensity of prion entity transmission to the

daughter cell partly depends on the size of the diffuse

oligomers In this model, larger aggregates are less

transmissible to the next generations, whereas smaller

oligomers are more transmissible Considering the

observation that Sup35 forms amyloid fibrils even in

cells, we depicted the size-dependency of the

transmis-sible propensity in Fig 4A, where the short amyloid

fibrils are represented as diffuse oligomers

As extreme cases, visible foci or rod⁄ ring-shaped

aggregates are barely transmissible By contrast, the

monomeric form of Sup35 is the easiest to transmit

Therefore, diffuse oligomeric states, which have

prop-erties distinct from those of the monomer in the

protein function, would be the appropriate size for the prion phenomena of protein-based inheritance

The behavior of diffuse oligomers of Sup35 in the GuHCl-treated [PSI+] cells can be explained by an impairment of the division phase by the inactivation of Hsp104 (depicted as the absence of the division phase in Fig 4B) Moreover, this scenario can be extrapolated to the size-dependent transmission of the prion aggregates, even in the presence of functional Hsp104 (Fig 4B)

A

B

Fig 4 Proposed model of size-dependent transmission of amyloid-based oligomers (A) Prion forms of Sup35 in [PSI + ] cells are composed of diffuse oligomers, which are basically amyloid fibrils fragmented in a variety of sizes The transmissible propensity is negatively correlated with the size: shorter fibrils are more trans-missible to daughter cells In extreme cases, visible spherical foci

or rod ( ⁄ ring)-shaped aggregates are barely transmissible By con-trast, the monomeric form of Sup35 is the easiest to transmit (B) Dynamics of oligomer remodeling in [PSI + ] cells are schemati-cally represented Upper cells are normal [PSI + ] cells, in which the growth (orange arrow) and division (green arrow) of amyloid-based oligomers are well balanced Smaller oligomers are preferentially transmitted to the next generations Overexpression of Sup35 often induces the formation of spherical foci, which exist in a dynamic equilibrium with diffuse oligomers Lower cells are Hsp104-inactivated [PSI + ] cells, in which the division phase is weakened (represented as the absence of the green arrow, for emphasis) Strong transmission bias in the cells eventually leads to the cure of [PSI + ] Overexpression of Sup35 in the cells often induces the formation of visible, rod-like aggregates.

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The proposal that there is a transmission bias, even in

the presence of functional Hsp104, might extend to

yeast prion ‘strains’ and other amyloid-forming protein

phenomena [PSI+] has multiple phenotypic strains,

including strong and weak [PSI+] [48–52] The fibrils

that cause the strong [PSI+] are known to be fragile,

resulting in the smaller size [51] Size-dependent

trans-mission explains why the strong phenotype is induced

by the smaller fibrils that tend to be transmitted to the

daughter In addition, a polyglutamine sequence fused

with the C-terminal domain of Sup35 forms amyloid

aggregates, but is not inherited as a prion [53,54] If the

polyglutamine aggregates were generally large, then

the preferential retention of the large aggregates in the

mother cells might result in the impaired inheritance

Regarding the transmission from mother cells to

daughter cells, the mechanism by which Sup35 proteins

are transmitted to daughter cells is unclear Is there an

energy-dependent transmission system, such as the

actin cytoskeleton? Alternatively, simple diffusion

might suffice for the transmission to the daughter cells

We can assume that a diffusion barrier exists at the

bud neck, where the transmission of large Sup35

oligo-mers is more or less restricted by an unknown

mecha-nism In fact, because the bud neck has diffusion

barriers, such as septin rings [55,56], this assumption

might be feasible

Implication to other prions

The importance of the diffuse oligomers in [PSI+]

yeast cells could be extended to other yeast prions as

well as to mammalian prions In fact, the dynamic

nat-ure of the visible foci is not restricted to Sup35

aggre-gates in [PSI+] cells Single-cell imaging revealed that

foci derived from Rnq1–GFP in [RNQ1], a yeast prion,

also disappeared during cell growth [36], suggesting

that foci derived from other prions besides those

formed by the Sup35 and Rnq1 proteins are dynamic

during their propagation In another yeast prion

[URE3], soluble forms of the [URE3] determinant Ure2

protein were linked to the [URE3] phenotype [57],

sug-gesting that the diffuse oligomers are critical for

main-taining the [URE3] prion Regarding mammalian

prions, the physiological relevance of PrP oligomers

remains to be identified However, we note that recent

studies have shown that PrP can also form soluble

oligomeric states [58–60]

Concluding remarks

Yeast prions are not toxic amyloids Instead, amyloid

forms are utilized to switch the functional state of a

protein Typically, the monomeric form is active, whereas the aggregated (amyloid) form is inactive, which is the molecular basis of the prion phenotypes

To maintain the phenotype from generation to genera-tion, that is, the protein-based inheritance, the amyloid structures must propagate For the propagation, the amyloid fibril, an ordered aggregate, has adopted a growth-and-division strategy for the protein switch, leading to the dynamic remodeling of the diffuse oligo-mers In this context, the amyloids that are used in the prion should be fragile Understanding, in greater detail, the intrinsic fragility of the amyloids and their susceptibility to trans-acting factors, such as Hsp104, will provide important insights into prion biology as well as into other amyloid-forming proteins in the cell

Acknowledgements

We thank A Kishimoto for Figure 1 This work was supported by Grants-in-Aid for Scientific Research (B) and on Priority Areas (17370034, 18031007, 19058002

to H.T.) from JSPS and MEXT, Japan

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