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In this review, we summarize the most recent work on calpain-dependent apoptotic neuronal cell death and the regulation of intracellular pathways involving calpain which may lead to neur

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Implication of calpain in neuronal apoptosis

A possible regulation of Alzheimer’s disease

F Raynaud and A Marcilhac

UMR5539, EPHE-CNRS-UM2, cc107, Universite´ de Montpellier II, France

Introduction

In recent years, enormous efforts have been made to

clarify the apoptotic pathways involved in neuronal

cell death Indeed, programmed cell death, or

apopto-sis, is beneficial during embryonic development and

adult life, but its dysregulation accompanies the

patho-genesis of many diseases Calpain appears to play an

important role in apoptosis in many cases, as discerned

by its up-regulation and the blockade of apoptosis by

calpain inhibitors

In this review, we summarize the most recent work

on calpain-dependent apoptotic neuronal cell death and the regulation of intracellular pathways involving calpain which may lead to neurodegenerative patholo-gies such as Alzheimer’s disease (AD)

Calpain and the regulation of apoptosis Proteolytic enzymes of the caspase family play a cen-tral role in initiating and sustaining the biochemical events that result in apoptotic cell death In some

Keywords

Alzheimer’s disease; apoptosis; calpain;

neurodegenerative disease; neuron

Correspondence

A Marcilhac, UMR5539, EPHE-CNRS-UM2,

cc107, Universite´ de Montpellier II, place E.

Bataillon, 34095 Montpellier cedex 5, France

Fax: +33 0467144727

Tel: +33 0467144775

E-mail: marcilhac@univ-montp2.fr

(Received 23 March 2006, accepted 31 May

2006)

doi:10.1111/j.1742-4658.2006.05352.x

Apoptotic neuronal cell death is the cardinal feature of aging and neurode-generative diseases, but its mechanisms remain obscure Caspases, members

of the cysteine protease family, are known to be critical effectors in central nervous system cellular apoptosis More recently, the calcium-dependent proteases, calpains, have been implicated in cellular apoptotic processes Indeed, several members of the Bcl-2 family of cell death regulators, nuc-lear transcription factors (p53) and caspases themselves are processed by calpains Progressive regional loss of neurons underlies the irreversible pathogenesis of various neurodegenerative diseases such as Alzheimer’s dis-ease in adult brain Alzheimer’s disdis-ease is characterized by extracellular plaques of amyloid–b peptide aggregates and intracellular neurofibrillary tangles composed of hyperphosphorylated tau leading to apoptotic cell death In this review, we summarize the arguments showing that calpains modulate processes that govern the function and metabolism of these two key proteins in the pathogenesis of Alzheimer’s disease To conclude, this article reviews our understanding of calpain-dependent apoptotic neuronal cell death and the ability of these proteases to regulate intracellular signa-ling pathways leading to chronic neurodegenerative disorders such as Alz-heimer’s disease Further research on these calpain-dependent mechanisms which promote or prevent cell apoptosis should help us to develop new approaches for preventing and treating neurodegenerative disorders

Abbreviations

AD, Alzheimer’s disease; AIF, apoptosis-inducing factor; APP, amyloid precursor protein; CaMKIV, calmodulin-dependent protein kinase type IV; cdk5, cyclin-dependent kinase 5; MAP, microtubule-associated protein; NMDAR, N-methyl- D -aspartate receptor.

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forms of apoptosis, the extrinsic apoptotic pathway is

initiated by activation of caspase 8 after death receptor

ligation In other forms, activation of the intrinsic

apoptotic pathway is initiated by caspase 9 and is

trig-gered by cytochrome c release from mitochondria [1]

This process is critically regulated by Bcl-2 family

pro-teins These pathways converge to activation of the

executioner caspases (e.g caspase 3)

Calpains and the Bcl-2 family

Members of the Bcl-2 family of proteins either promote

or repress programmed cell death [2] Several members

are processed by calpains [3] Using the model of

trophic factor deprivation in sympathetic neurons, it

has been shown that Bax translocation from the cytosol

to the mitochondria is a critical event in neuronal

apoptosis [4] In this context, calpain cleaves Bax into a

pro-apoptotic 18-kDa fragment which promotes

cyto-chrome c release and apoptosis [5]

Moreover, cleavage of Bid (another pro-apoptotic

Bcl-2 family member) by calpain has been implicated

in mitochondrial permeabilization and cell death

fol-lowing ischemia⁄ reperfusion in the heart [6] Indeed,

truncated Bid induced cytochrome c release from brain

mitochondria and apoptosis-inducing factor (AIF)

release only in the presence of active calpain AIF has

been shown to translocate from mitochondria to the

cytosol as well as the nucleus when apoptosis is

induced In a recent study, Polster et al [7] suggested a

novel mechanism of AIF release that is mediated by

direct proteolysis of the protein by calpain, removing

its association with the mitochondrial inner membrane

They proposed an experimental scheme in which the

calpain 1 cleaves Bid into a more active form in order

to permeabilize mitochondrial outer membrane and

allow calpain access to the intermembrane space

Cal-pain 1 then cleaves AIF, releasing truncated AIF,

which could regulate apoptosis So AIF is a novel

cal-pain substrate that has been implicated in neuronal cell

death [7] (Fig 1)

Cross-talk between calpains and caspases

The striking similarity between the substrates for

casp-ases and calpains raises the possibility that both

prote-ase families contribute to structural dysregulation and

functional loss of nerve cells under neurodegenerative

conditions Cross-talk between calpains and caspases

has been reported during apoptosis of neuronal cells

induced by a prion protein fragment [8] Moreover,

calpains are able to be activated via caspase-mediated

cleavage of calpastatin during initiation of apoptotic

execution [9], and the disturbance of intracellular cal-cium storage associated with ischemic injury may induce apoptosis through calpain-mediated caspase 12 activation and Bcl-xL inactivation [10] (Fig 1) Although calpains may enhance caspase activity, they can also function to block the activation of caspases For example, calpains can cleave caspase 9 rendering it incapable of activating caspase 3 and preventing the subsequent release of cytochrome c [11] Yamashima [12] added to this dual cross-talk another effective can-didate, cathepsins, which are implicated in neuronal cell death He suggested a possible cascade of events involving three protease systems: calpain-induced cath-epsin release, cathcath-epsin-mediated caspase activation and caspase-mediated calpastatin degradation leading

to enhancement of calpain activity

Calpains and transcription factor regulation DNA damage is an initiator of neuronal death impli-cated in neuropathological conditions such as stroke Previous evidence has shown that apoptotic death of embryonic cortical neurons treated with the DNA-damaging agent camptothecin is dependent on the tumor suppressor p53, an upstream death mediator, and more distal death effectors such as caspases Calpains can act as an alternative system to the pro-teasome in regulating the stability of p53 family mem-bers Several recent reports have highlighted a possible role for calpains in the cleavage of p53 In particular, Kubbutat & Vousden [13] have shown that a pre-ferential site for calpain cleavage exists within the N-terminus of p53 Calpain inhibition leads to p53 sta-bilization and to altered cell cycle progression Both calpain 1 and 2 can cleave p53 with a different degree

of susceptibility to cleavage in various p53 mutants The cleavage of p53 by calpains can occur under pathological conditions and contributes to the DNA damage response [14] More recently, Munarriz et al [15] have shown that p73, another component of the DNA damage response, which belongs to the family of transcription factors that includes p53 and p63, is also

a substrate for calpains In this case, calpain regulation can control the steady-state protein concentration of different isoforms of p73, and calpain-mediated degra-dation of p73 may have a regulatory, physiological function, in addition to a potential role in cell death

So, several nuclear transcription factors are calpain substrates leading to the suggestion that calpains can regulate transcriptional events

Moreover, translocation of calpain to the nucleus may play a role in apoptosis For example, Tremper-Wells

& Vallano [16] have demonstrated in dissociated

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cultures of cerebellar granule cells that

calpain-mediated Ca2+⁄ calmodulin-dependent protein kinase

type IV (CaMKIV) proteolysis is an autoregulatory

feedback response to sustained activation of a

Ca2+⁄ CaMKIV signaling pathway (CaMKIV mediates

phosphorylation of different transcription factors such

as CREB and regulates expression of prosurvival⁄

antiapoptosis genes.)

Calpains, glutamate receptor and NF-jB

The excitatory neurotransmitter glutamate is a key

player in neuronal plasticity, development and

neuro-degeneration Stimulation of glutamate receptors

[N-methyl-d-aspartate receptors (NMDARs)] leads

to Ca2+-mediated apoptosis, a response that may

contribute to excitatory neuronal toxicity Calpain

activation in neurons has been predominantly linked

to cell death during ischemia and stroke [17–19] How-ever, the literature is contradictory on this subject Indeed, recent results show that prolonged activation

of NMDARs in neurons activates calpain, and activa-ted calpain in turn down-regulates the function of NMDARs, which provides a neuroprotective mechan-ism against NMDAR overstimulation accompanying ischemia and stroke [20] Scholzke et al [21] have shown that glutamate activates NF-jB through calpain

in neurons Moreover, after glutamate exposure, the specific calpain inhibitor, calpeptin, prevents IjBa degradation and therefore NF-jB activation However, the role of IjBa degradation by calpain in glutamate-induced cell death is difficult to predict as both proapoptotic and antiapoptotic effects have been attributed to NF-jB [22,23]

Fig 1 Scheme illustrating the regulation of

Bcl-2 family proteins, cytochrome c release

and apoptosis by calpains (1) Cleavage of

Bax by calpain, formation of truncated Bax

(tBax) and stimulation of cytochrome c

release; (2) cleavage of Bid by calpain and

formation of truncated Bid (tBid) leading to

(2a) direct release of cytochrome c and ⁄ or

(2b) permeabilization of mitochondrial outer

membrane, translocation of calpain in the

intermembrane space and AIF cleavage

(tAIF) and release; (3) calpain-mediated

Bcl-xL inactivation and cytochrome c

release; (4) calpain-mediated p53 activation:

p53 induces Bcl-xL inactivation, Bax

stimulation and cytochrome c release.

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Astrocytes contribute to the neuroprotection and

survival of neurons: any astrocytic dysfunction

seri-ously affects neuronal viability The study of astrocytes

is particularly important, considering the coexistence

of the apoptotic death of neurons and astrocytes in

damaged brains suffering from ischemia and

neurode-generative diseases Calpain inhibitors,

Leu-norleucinal (calpain inhibitor 1) and

N-acetyl-Leu-Leu-methioninal (calpain inhibitor 2), decrease Ca2+

reperfusion-induced H2O2 production and apoptotic

cell death in cultured rat astrocytes These findings

suggest that calpain is involved in Ca2+-mediated

apoptosis in astrocytes In this model of astrocyte

apoptosis, translocation of the NF-kB p65 subunit to

the nucleus is observed These findings indicate that, in

this case, NF-jB acts as a death-promoting factor in

apoptosis of cultured astrocytes [24]

Calpain and Alzheimer’s

neuro-degenerative disease

Neuronal cell death in acute and neurodegenerative

disorders occurs by a variety of biochemical and

mor-phological alterations [25]

AD leads to a progressive deterioration of cognitive

function with loss of memory Neuronal injury

pre-sents in the region of the brain containing the

hippo-campus and the cortex AD is characterized by two

pathological hallmarks consisting of extracellular

plaques of amyloid–b peptide aggregates [26] and

intracellular neurofibrillary tangles composed of

hyperphosphorylated microtubular protein tau [27]

The b-amyloid deposition that constitutes the plaques

is composed of a 39–42 amino-acid peptide (Ab),

which is the proteolytic product of the amyloid

precur-sor protein (APP) by b⁄ c secretases Calpains

modu-late processes that govern the function and metabolism

of key proteins in the pathogenesis of AD including

tau and APP [28]

Calpains in the brains of patients with AD

Calpains are known to regulate the activities of various

enzymes, including several protein kinases and

phos-phatases that modify the cytoskeleton in addition to

direct cleavage of cytoskeletal proteins (Lebart &

Benyamin, [28a]) Alterations in calcium homeostasis

in AD pathogenesis [29] associated with calpain

over-activation [30] have been proposed to play an

import-ant role in the development of cytoskeletal pathology

and neurodegeneration Calpain activation has been

found in clinical brain specimens of AD For example,

calpain 2 was demonstrated to be present in 75% of

neurofibrillary tangles [31] Moreover, calpain 1 is acti-vated, which in turn cleaves and activates calcineurin

in the AD brain and this phenomenon correlates with the number of neurofibrillary tangles [32] Calpastatin,

a specific calpain inhibitor, is altered in AD Indeed, it decreases as the number of plaques and tangles increase in AD brains [33] Moreover, calpain inhibi-tors were able to restore normal cognition and synap-tic transmission in a transgenic model of AD [34] Mutations in PS1 (presenilin) that cause early onset familial AD can sensitize cells to DNA-damage-induced death and increase the production of Ab In hippocampal neurons expressing mutant PS1, the hypersensitivity to DNA damage correlates with increased intracellular calcium concentrations, up-regu-lation of calpain 1, and induction of p53, leading to neuronal apoptosis [35] Moreover, calpains 1 and 2 have been shown to regulate PS1 activity by direct cleavage of this protein [36]

Calpains and amyloid formations in AD Different data support the hypothesis that calpains are involved in the alpha cleavage of APP in vivo and thus are able to stimulate the nonamyloidogenic pathway, leading to a decrease in Ab42 release

Indeed, APP is cleaved within its Ab region by a-secretase to release a soluble N-terminal fragment (denoted sAPP) A number of studies have shown that enhancing a-secretase activity can reduce Ab produc-tion APP-a processing is sensitive to a variety of regulatory agents such as phorbol esters, glutamate, calcium ionophore For example, some authors demon-strated that the stimulated sAPP release by phorbol esters involves calpain activation in the cells, suggesting that calpain, particularly calpain 1, is a potential candi-date for an a-secretase substrate in the regulated APP a-processing [37,38] These results support the hypothe-sis of a decrease in calpain activity during aging leading

to selective accumulation of its substrates (e.g APP) and an increase in Ab42 production in cultured cells [39] Consistent with this, it has been reported that infusion of calpain inhibitors into the rat brain results

in accumulation of Ab or Ab-containing fragments [40] This effect is not unexpected because calpain is essential for life, and severe inhibition of its activity could be harmful to cells Although this novel model for plaque formation is in agreement with both the property of calpain that consists of specific and limited cleavage of target proteins, and data that show that other calpain susbtrates such as spectrin (fragment) are also deposited during aging [41], this model remains controversial and awaits additional experimental tests

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Indeed, although there is evidence in favor of

cal-pains acting as a-secretases, such as (a) the a-secretase

cleavage site is identical with a cleavage site of calpain

in protein kinase C, (b) calpains are colocalized with

APP in situ in different structures including neurons,

astroglia, senile plaques, and synapses, (c) some

rea-gents that enhance a-secretase activity are well-known

calpain activators, and (d) considering its vulnerability

to oxidative stress, a-secretase may belong to the

fam-ily of cysteine proteases, a serious problem with this is

that calpains are intracellular and may be not able to

reach the cleavage site of APP, which is located

out-side of the membrane However, in support of calpains

acting as secretases is the fact that these proteases are

involved in cleavage of other membrane surface

pro-teins, including growth factor receptors, integrin and

glutamate receptors, leading to the release of their

extracellular domains [42]

During the extended time course of AD in humans,

the calpain system plays multiple roles Indeed, if

reduced calpain activity were to promote AD

pathol-ogy by increasing Ab42 generation, this would contrast

with increased calpain activity promoting pathological

changes in tau

Calpains and tau regulation in AD

Based on a growing literature, cyclin-dependent kinase

5 (cdk5), which promote phosphorylation of tau, has

been implicated in the pathological processes that

contribute to neurodegeneration in AD p35 is a

neuron-specific activator of cdk5, and conversion of

p35 into p25 by calpain-dependent proteolysis causes

prolonged activation and mislocalization of cdk5

Con-sequently, the p25⁄ cdk5 kinase hyperphosphorylates

tau, disrupts the cytoskeleton, and promotes apoptosis

of primary neurons Application of the

amyloid–b-pep-tide(1–42) induces the conversion of p35 into p25 in

neurons, and inhibition of cdk5 or calpain activity

reduces cell death in these conditions [43,44]

More-over, a recent study showed that preaggregated Ab

induced the generation of a neurotoxic 17-kDa tau

fragment, which is prevented by a calpain inhibitor in

cultured hippocampal neurons [45] This proteolytic

cleavage may lead to neurite degeneration by reducing

the pool of full-length tau available for binding to

microtubules The decrease in tau bound to

microtu-bules could in turn reduce their stability and promote

a more rapid depolymerization cycle and therefore the

disruption of the microtubule network [45] Veeranna

et al [46] demonstrated that, under conditions of

cal-cium injury in neurons, calpains are upstream

activa-tors of Erk1,2 signaling and probably mediate, in part,

the hyperphosphorylation of neurofilaments and tau seen at early stages of AD

Besides the alteration of the structure and properties

of tau [the most studied member of the microtubule-associated protein (MAP) family], modifications of other members of this family (such as MAP1A, MAP1B and MAP2) may contribute to the perturba-tion of the microtubule network in AD and ultimately lead to neuronal degeneration without accumulation of amyloid deposits A recent study [47] showed that sol-uble Ab oligomers induce time-dependent degradation

of MAP1A, MAP1B and MAP2 Calpain activation is sufficient on its own to proteolyse MAP2a,b,c iso-forms, whereas MAP1A and MAP1B sequential pro-teolysis results from caspase 3 and calpain activation This work confirms the cross-talk between caspase and calpain and identifies a novel mechanism associated with the proteolysis of several MAPs and leading to neuronal apoptosis in AD

Conclusion

In summary, the ubiquitous expression of calpains in distinct subcellular compartments at different matura-tional stages and the diversity of substrates indicate that they are multifunctional effectors of myriad intra-cellular processes

Moreover, progressive cell loss in specific neuronal populations is a pathological hallmark of neurodegen-erative diseases, and calpain is a Ca2+-activated pro-teolytic enzyme involved in neurodegeneration in a variety of injuries and diseases of the central nervous system

Thus, identification of mechanisms that involve cal-pains and either promote or prevent cell apoptosis provides new approaches for treating diverse neuro-degenerative disorders including AD

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