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Tiêu đề DNA methylation-mediated nucleosome dynamics and oncogenic Ras signaling: insights from FAS, FAS ligand and RASSF1A
Tác giả Samir K. Patra, Moshe Szyf
Trường học McGill University
Chuyên ngành Cancer epigenetics
Thể loại Review article
Năm xuất bản 2008
Thành phố Montreal
Định dạng
Số trang 19
Dung lượng 522,26 KB

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Abbreviations aSMAase, acid sphingomyelinase; DISC, death-inducing signaling complex; DNMTs, DNA methyltransferases; FADD, FAS-associated death domain; FASL, FAS ligand; gld, generalized

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DNA methylation-mediated nucleosome dynamics and

oncogenic Ras signaling

Insights from FAS, FAS ligand and RASSF1A

Samir K Patra1,* and Moshe Szyf2

1 Cancer Epigenetics Research, Kalyani, India

2 Department of Pharmacology and Therapeutics, McGill University, Montreal, Canada

DNA methylation and chromatin modification and

remodeling are currently center stage in studies of the

epigenetic regulation of genome function in normal

physiology, disease states and development [1–25]

Sev-eral isoforms of enzymes catalyzing both DNA and

histone modifications have been characterized

Con-comitant with differentiation, cell-type-specific patterns

of DNA methylation and histone modification are

gen-erated and are believed to program cell-type-specific

physiological functions, including memory formation

in neurons [2,18,20] These elaborate epigenetic programs may be difficult to reverse and rebuild during animal cloning procedures, because the signals and mechanisms for gene-specific hypermethylation and global demethylation patterns are not completely understood [2] In eukaryotes, the chromatin is orga-nized as euchromatin and heterochromatin Euchroma-tin encompasses the majority of single-copy genes, it replicates during early S phase and contains acetylated histones Heterochromatin is composed of long

Keywords

apoptosis; cancer; DNA methylation;

epigenetics; FAS; FAS ligand; H-Ras; K-Ras;

nucleosome dynamics; RASSF1A

Correspondence

S K Patra, Cancer Epigenetics Research,

Kalyani (B-7 ⁄ 183), Nadia, West Bengal, India

Fax: +91 332 582 8460

Tel: +91 943 206 0602

E-mail: skpatra_99@yahoo.com

*Present address

Division of Biochemistry, Department of

Experimental Medicine, University of Parma,

Italy

(Received 5 June 2008, revised 8 August

2008, accepted 22 August 2008)

doi:10.1111/j.1742-4658.2008.06658.x

Cytosine methylation at the 5-carbon position is the only known stable base modification found in the mammalian genome The organization and modification of chromatin is a key factor in programming gene expression patterns Recent findings suggest that DNA methylation at the junction of transcription initiation and elongation plays a critical role in suppression

of transcription This effect is mechanistically mediated by the state of chromatin modification DNA methylation attracts binding of methyl-CpG-binding domain proteins that trigger repression of transcription, whereas DNA demethylation facilitates transcription activation Under-standing the rules that guide differential gene expression, as well as tran-scription dynamics and transcript abundance, has proven to be a taxing problem for molecular biologists and oncologists alike The use of novel molecular modeling methods is providing exciting insights into the chal-lenging problem of how methylation mediates chromatin dynamics New data implicate lipid rafts as the coordinators of signals emanating from the cell membrane and are converging on the mechanisms linking DNA meth-ylation and chromatin dynamics This review focuses on some of these recent advances and uses lipid-raft-facilitated Ras signaling as a paradigm for understanding DNA methylation, chromatin dynamics and apoptosis

Abbreviations

aSMAase, acid sphingomyelinase; DISC, death-inducing signaling complex; DNMTs, DNA methyltransferases; FADD, FAS-associated death domain; FASL, FAS ligand; gld, generalized lymphoproliferative disorder; lpr, lymphoproliferative disorder; MAPK, mitogen activated protein kinase; MBD, methyl-CpG-binding domain proteins; MGMT, O6-methylguanine methyltransferase; RESE, Ras epigenetic silencing effectors; TNF, tumor necrosis factor.

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stretches of DNA repeats, replicates in late S phase

and contains lower levels of acetylated histones and

higher levels of DNA methylation [1–3,5–7,9,13–17,21–

24] Cytosine methylation is implicated in controlling

transcription, maintaining genome stability, parental

imprinting and X chromosome inactivation [1,2,21,

24,25]

The DNA methylation-mediated repression of

sev-eral genes, including those encoding proteins involved

in cell-cycle regulation and apoptosis, is a major cause

of tumor development and cancer progression [1–9] In

addition to the gene-specific hypermethylation of

sev-eral genes in many cancers, genomes of tumor cells are

globally hypomethylated and several genes critical for

tumor metastasis and progression are activated by

demethylation [2,3,6,11–16] The enzymes and

cofac-tors responsible for demethylation in cancer cells are

currently unknown It is known, however, that during

early development asymmetric DNA demethylation of

the paternal genome is observed just after fertilization

[2,17–19] Similar to the global hypomethylation

observed in cancer, the enzymes responsible for this

demethylation are unknown

Although the DNA methylation pattern is

pro-grammed during development, it remains highly

sensi-tive to both the chemical and social environment [10]

Studies have suggested that bioactive components of

food, both essential and nonessential nutrients, can

modify DNA methylation patterns in complex ways

For example, consumption of a methyl-deficient diet

led to hypomethylation of specific CpG sites within

several oncogenes (such as c-myc, c-fos and c-H-Ras),

resulting in high expression of these genes [26] Recent

studies have shown that tea catechins are effective

inhibitors of human DNA methyltransferase

(DNMT)-mediated DNA methylation in vitro, and re-expression

of a few genes in cultured cancer cells is observed in

response to tea catechins [27,28] Thus, DNA

methyla-tion may be viewed as an interface between the

envi-ronment and the human genome It stands to reason

that it might play critical role in several human

pathol-ogies, in particular age-related disease

DNA methylation enzymes

In mammalian cells, DNA methylation is catalyzed

by two classes of DNMT DNA-methyltransferase-1

(DNMT1; EC 2.1.1.37) is essential for maintaining

DNA methylation patterns in proliferating cells and is

also involved in establishing new DNA methylation

patterns; de novo methylation Members of the second

class of methyltransferases, DNMT3a and DNMT3b

are required for de novo methylation during embryonic

development [2,25], whereas DNMT3L cooperates with the DNMT3 family to establish maternal imprints in mice [29] DNMT1 and DNMT3B interact among themselves [30] and DNMT3A interacts with histone methyltransferases SETDB1 in the promoters of silenced gene during cancer development [16] Catalytic mechanisms of DNMTs involve the formation of a covalent bond between a cysteine residue in the active site of the enzyme and carbon 6 (C6) of cytosine in DNA The mechanisms involved have been described recently [2,25,31–39] Very recent data suggest that DNMTs may also be involved in the deamination of methylated cytosines to thymines [31,32] The mis-matched thymidine is then removed by base⁄ nucleotide excision repair resulting in repair to an unmethylated cytosine [2] This has been proposed to serve as a mechanism for dynamic DNA methylation [31,32]

A different type of DNA methyltransferase is O6-methylguanine DNA methyltransferase (MGMT;

EC 2.1.1.63) This enzyme does not methylate DNA but is a DNA repair protein that removes mutagenic and cytotoxic adducts from the O6 position of guan-ine O6-Methylguanine often mispairs with thymine during replication Following DNA replication this would result in conversion of a guanine–cytosine (GC) pair to an adenine–thymine (AT) pair Thus, repairing O6-methylguanine adducts is essential for the integrity

of the genome Interestingly this DNA methyltransfer-ase is regulated by DNA methylation Hypermethyla-tion of the MGMT promoter is associated with loss of MGMT expression⁄ function in many tumor types [1,4,40] MGMT hypermethylation is an example of the emerging field of pharmacoepigenomics The impact of chemotherapy would be dependent on the epigenetic state of cardinal genes such as MGMT [1,41] Knowing the state of methylation of critical repair genes is critical for the proper planning of a chemotherapeutic protocol

Removal of the methyl group (MeC-DNA demethy-lation) from critical positions in promoters is essential for the transcription of many genes A long line of evidence suggests that active enzymatic MeC-DNA demethylation occurs in nonreplicating cells to induce the transcription of specific genes at distinct time points [2] The mechanisms of demethylation are unknown and the enzymes involved are not firmly established One possible mechanism is through forma-tion of a cytosine–Michael adduct⁄ complex with MBD2 protein but this would require a cofactor [2,42– 48] which is not known The current notion of Michael adduct chemistry is that in such types of complex the SN2 mechanism would not occur In principle, water added across the 4C–5C double bond with the

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hydroxyl group attacking carbon 4, followed by

elimi-nation of ammonia will yield thymidine [2,25,34,46]

Epigenetic consequences of DNA

modifications – nucleosome dynamics

There is a bilateral relationship between DNA

methyl-ation and chromatin structure [2,16,49–58] Promoters

of genes and important regulatory sequences are

asso-ciated with hyperacetylated histones, whereas silent

genes are associated with hypoacetylated histones

Acetylated histones are associated with unmethylated

DNA and are rarely present in methylated DNA

regions [59] In addition to histone acetylation, which

plays a critical role in gene regulation, other histone

modifications such as methylation, phosphorylation

and ubiquitination play a similar role in regulating

genome functions [49–56] A combinatorial

arrange-ment of these modifications is believed to constitute a

‘histone code’ Methylation of DNA and deacetylation

of histones H3 and H4, combined with methylation of

K27 residue on the H3-histone tail in upstream

regula-tory regions leads to inactivation⁄ repression of gene

expression, whereas selective acetylation of histones

H1, H3, H4, methylation of H3K4 and DNA

deme-thylation are associated with activation of

transcrip-tion [2,5,6,12,16,22,23] (Fig 1)

How does DNA methylation signal for repression of

transcription? Repression of transcription may occur

through different mechanisms One simple mechanism

is that DNA methylation interferes with the binding of

transcriptional activators [24,25,53,55,59] A second

mechanism involves recruitment of

methyl-CpG-bind-ing domain proteins (MBDs), such as MBD1, MBD2,

MBD3, MBD4 and MeCP2 MBDs recruit

co-repres-sor complexes to methylated genes, which include

histone-modifying enzymes such as histone

deacetylas-es and histone methyltransferasdeacetylas-es precipitating an

inac-tive chromatin structure [16,21,39,49,51,56] This

mechanism provides an explanation for the correlation

between DNA methylation and inactive chromatin

configuration

DNA methylation and histone modification act in

concert to program gene expression Figure 1 presents

a model of the inhibition of gene expression by DNA

methylation Cytosine methylation at the DNA

sequence d(GGCGCC)2 triggers an extended eccentric

double-helix structure called E-DNA Like B-DNA,

E-DNA has a long helical rise and the base is

perpen-dicular to the helix axis The 3¢-endo sugar

conforma-tion provides the characteristic deep major groove and

shallow minor groove of A-DNA [60] Analysis of the

hydration pattern around methylated CpG sites in

crystal structures of A-DNA decamers at three high resolutions (1.7, 2.15 and 2.2 A˚) reveals that the methyl groups of cytosine residues are well hydrated with a higher amount of Mg2+ in their vicinity [61], which facilitates the interaction of MBD proteins and chromatin remodeling machines with the MeCpG sites The MBD–MeCpG complex then brings about deacety-lation of histones H3 and H4 [2,22] by recruiting class I histone deacetylases, which may be co-recruited with DNA-topoisomerase II [62] (Fig 1) Indeed, it has been shown experimentally that methylation of DNA brings about general deacetylation of histones H3 and H4, prevents methylation at H3K4 and induces methylation of H3 K9 [2,52–56] Histone H3K4 trimethylation is associated with transcription-ally active genes [59,63–73] MeCP2 has also been shown to recruit the histone methyltransfaerase SUV39 which targets H3 K9 [74] Okitsu & Hsieh observed a tight correlation between depletion of H3K4Me2 and regions of DNA methylation, and pro-posed that DNA methylation dictates a closed chroma-tin structure devoid of H3K4Me2 [59] The recent discovery of histone demethylases has challenged the originally held belief that histone methylation is static Histone demethylases specific for mono-, di- and trimethylated histone H3K4 are now known and their structures have been described [54,70–73,75–78] It is possible that the recently discovered histone demethy-lase LSD1 also participates in maintaining methylated regions of DNA devoid of H3K4 methylation [56,78] DNA methylation immediately downstream of the transcription start site has a dramatic impact on tran-scription, affecting transcription elongation rather than initiation Recent findings suggest that DNA methyla-tion at the juncmethyla-tion of transcripmethyla-tion initiamethyla-tion and elongation is most critical in transcription suppression and this effect is mechanistically mediated through chromatin structure [53,56,59,78] Although some important ideas have been suggested in other studies [64,68–70], it is still difficult to predict the effect of methylated DNA segments on transcription because differences in the size and position of the methylated DNA regions may differentially affect transcription For example, although a methylated coding region positioned 1 kb downstream of the promoter has little impact on transcription initiation, as observed by Lorincz et al [65], the same methylated sequence might have a much larger impact on transcription initiation if positioned immediately downstream of the promoter [54,64,68–70,78] The context seems to be critical [56] Recent data suggest that not only can DNA methyl-ation direct the formmethyl-ation of inactive chromatin struc-ture, but also that histone signals can direct DNA

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Fig 1 Cytosine base in DNA – the amazing switch for the regulation of gene expression and chromatin remodeling Cytosine, extended from the sugar-phosphate backbone (black circles–pink lines) and expanded manifold beyond the scale, is the only base in mammalian chro-mosomes which is stably modified by methylation at the carbon-5 position (formation of - Me CpG-) after replication DNA methylation inhibits gene expression affecting chromatin structure [2,6,22,59–71], because the presence of methyl groups on DNA affect the structure of DNA and the interaction of other proteins and enzymes with local nucleosomes [2,60] Methylation of DNA (MeCpG-) brings about a general hydra-tion of DNA [61], which facilitates the methyl-CpG-sequence binding (MBD) proteins to recognize the Me CpG- sites in nucleosomes for remodeling into a repressive complex DNMT- Me CpG- influence deacetylation of histones H3 and H4 by recruiting class I histone deacety-lases (HDACs); prevents methylation at H3K4, and induce methylation of H3 K9 in eukaryotes [56–69] HDACs may be co-recruited with DNA-topoisomerase II [62] Histone H3K4 methylation is associated with transcriptionally active nucleosomes of chromatin in which K4 of H3 are trimethylated, whereas H3 K27 methylation is associated with inactive chromatin [56,59,63–68,79] Methylation of histones is revers-ible and histone demethylases specific for di- and trimethylated histone H3K4 are discovered; for example, LSD1 represses transcription through demethylation of H3K4 Me3 [72,75,77] Okitsu & Hsieh [59] observed a tight correlation between the depletion of H3K4Me2 in the regions of DNA methylation Conversely, the level of H3K4Me2 remains high in the unmethylated DNA regions regardless of the presence

of RNA Pol II It can be proposed that Me CpG- dictates a closed chromatin structure that is devoid of H3K4Me2 and inhibits transcription, and that the presence of H3K4me2 marks an open chromatin structure that would permit transcription if all other conditions for active tran-scription are fulfilled [56,58] In early development, genomic methylation is erased and the somatic methylation pattern is re-established at the time of implantation The initiation of DNA demethylation-dependent nuclear processes is highly dependent on unfolding of chromatin structure In this context, acetylation of lysine ⁄ arginine of histone tails of H3 and ⁄ or H4 at the respective Me

CpG-rich nucleosome depends

on histone acetyl transferases (HATs) [48–50] In addition to methylation, H3 K9, H3 K14, H3 K23 and H3 K27 are also prone to acetylation, whereas as H3 K18 is only acetylated [22,49–59,63–69] This implies that nucleosome position is biased by the DNA sequence to facilitate access to initiation factors and activators by hundreds of histone modification (deacetylation, demethylation and also phosphorylations at ser-ine ⁄ threonine residues) Also, activation of specialized domains by removal of loosely associated mobile proteins, including HMG, HP and H1, partly regulates the expression of independent genes modulating the access of the above factors [2,22] Note: all the modifications men-tioned here are not require for activation of a particular type of gene Histone decoding, DNA modifications and the accessory factors are predominantly dependent on the types of signals a cell receives for activation ⁄ repression of a specific gene or for a particular class of gene.

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methylation For example, methylation of tumor

sup-pressor genes in cancer usually occurs in regions of

DNA associated with H3-histone K27 methylation, a

suppressive histone mark [2,12,22,54] The histone

methyltransferase EZH2 recruits the DNA

methyl-transferase to targets of EZH2 in the genome [1]

Thus, there is a bilateral relationship between DNA

methylation and inactive chromatin configuration

DNA demethylation is also tightly associated with

chromatin structure; histone acetylation of H3 and H4

histone tails is a hallmark of active chromatin

configu-ration and transcriptionally active regions of the

gen-ome [22,48,52,53,55,57–59] Hypgen-omethylation of DNA

[2,11,14] is found in regions associated with

hyperacet-ylated histones, and pharmacological histone

acetyla-tion could induce DNA demethylaacetyla-tion [48,79–81]

Thus, DNA demethylation, like DNA

hypermethyla-tion, has a bilateral relationship with chromatin

modi-fication

Ras oncogenes and oncoproteins

Ras, Rho, Rab, Arf and Ran are the five major classes

of monomeric GTPases whose biological functions are

regulated by the Ras family GTPases The cellular Ras

oncogene encodes a 21-kDa guanine

nucleotide-bind-ing protein, which plays a role in the regulation of

growth and differentiation in eukaryotic cells [1,82]

Despite profound improvements in our understanding

of the molecular and cellular mechanisms of action of

the Ras proteins, the expanding list of downstream

effectors and the complexity of the signaling cascades

that they regulate suggest that much remains to be

learnt [83] The study of Ras proteins and their

func-tions in cell physiology has led to many insights not

only into tumorigenesis but also into many

develop-mental disorders [82–84]

Although Ras binds both GDP and GTP with very

high affinity, the GTP-bound form is active and the

GDP-bound form is inactive The rate of intrinsic

nucleotide exchange and GTPase activity is very slow

Ras–GDP predominates in resting cells, but when

Ras is activated, specific guanine nucleotide exchange

factors enhance nucleotide exchange, increasing the

Ras–GTP complex Ras–GTP then activates

down-stream effectors such as Raf-1 GTPase-activating

pro-tein, however, causes the precipitation and

accumulation of inactive Ras–GDP within a cell and

may deregulate cellular physiology when overexpressed

[1,82–90] The retroviral oncogene, V-Ras, encodes a

protein that differs from the C-Ras product by a point

mutation that maintains this Ras protein constitutively

active [83] The Ras-related GTPase, Rho is required

for transmission of a proliferative signal by Ras If Rho is inhibited, the constitutively active Ras induces the cyclin-dependent kinase inhibitor p21(Waf1)⁄ Cip1, which blocks entry into the DNA synthesis phase of the cell cycle Rho activity suppresses induction of p21(Waf1)⁄ Cip1 by Ras, thus overcoming the block on entry into the S phase of the cell cycle Cells lacking p21(Waf1)⁄ Cip1 activity do not require Rho for the induction of DNA synthesis by activated Ras [1,83]

Lipid rafts and Ras signaling

Palmitoylation of N-Ras, K-Ras-4A and H-Ras, but not K-Ras-4B, in their C-terminal hypervariable regions is commonly required for their membrane relo-cation After relocation to the membrane, H-Ras and K-Ras-4A are translocated to lipid rafts; however, K-Ras-4B remains in the non-raft portion of the mem-brane [83] Hypervariable regions are responsible for targeting the isoforms to different microdomains in membrane Ras proteins localize to different plasma membrane microdomains, lipid rafts, formed by segre-gation of lipids based on their dissimilar biophysical properties [83,91] A comprehensive model of how Ras proteins are clustered for amplification, internalized and transmit their signals has recently been proposed [1] Eisenberg et al [92], employing fluorescence recov-ery after photobleaching, demonstrated coupling between membrane domains (rafts) in the external and internal leaflets of the plasma membrane and showed that this coupling modulated transbilayer signal trans-duction

Ras circulates between the Golgi, the endoplasmic reticulum and the plasma membranes H-Ras local-ized in the membranes of the endoplasmic reticulum and Golgi apparatus is activated by epidermal growth factor [1,91–93] After post-translational modification

in the endoplasmic reticulum, K-Ras is transported to the plasma membrane by a desorption–absortion mechanism [92,94] Ras detachment from lipid rafts requires GTP hydrolysis [92,93] H-Ras and N-Ras are transported to different sub-compartments by vesicular traffic, or by a nonvesicular pathway involv-ing a constitutive deacylation–reacylation cycle [1,83,92,95–97]

Inter-relationship of genetics and epigenetics in Ras oncogenic signaling

There is a bilateral relationship between genetic and epigenetic mechanisms in the activation of oncogenic Ras signaling Activation of K-Ras and H-Ras in human cancers results in DNA hypermethylation

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of target genes [1,6,31,83,98] Epigenetic

deregula-tion of critical repair genes can, however, increase the

rate of mutation of Ras genes (Fig 2) For example,

silencing of the repair methyltransferase MGMT would

result in an increased rate of mutation of Ras and

other oncogenes Figure 2 represents a scheme of how

the loss of MGMT expression would result in G to

A transitions in the K-Ras oncogene and in p53

[1,41,99] Indeed, MGMT promoter hypermethylation

is significantly pronounced in tumours that also bear a

G to A mutation in p53 suggesting a link between

epigenetic and genetic events and apoptosis related

diseases [99]

Genetic activation of Ras would also cause a change

in the state of methylation of several genes

Constitu-tive activation of Ras induces DNMT1 expression at

the transcriptional level through activation of cJUN

[100–102] The excess of unscheduled DNMT levels

would target certain genes for hypermethylation It is

believed that promoters marked by H3-K27

methyla-tion are targets of hypermethylamethyla-tion perhaps through

recruitment of DNMTs by EZH2 [103,104] Indeed,

targeting the Ras signaling pathway by drugs such as

methotrexate and inhibitors of ERK⁄ mitogen activated

protein kinase (MAPK) decreases DNA methylation in

malignant hematologic diseases and colon cancer cells indicating a causal relationship between Ras signaling and DNA methylation [105–108]

Ras activation would affect the DNA-methylation state and chromatin dynamics in the other direction as well Expression of v-H-Ras in mouse embryonal P19 cells resulted in genome-wide demethylation of certain genes, including a skeletal muscle-specific gene, adrenal cortex (c21)-specific gene, ubiquitous genes and exo-genously introduced sequences [100,109] Hence, DNA demethylase might be a potential downstream effector

of Ras signaling [1,2] Also, stimulation of the Ras– MAPK pathway leads to chromatin modification by histone H3 serine 10 and 28 phosphorylation in an acetylation-dependent and -independent fashion [55]

In summary, activation of the Ras-signaling pathway would trigger the methylation aberrations and histone modifications that are a hallmark of cancer: regional DNA hypermethylation and global hypomethylation

An attractive hypothesis is that K-Ras or H-Ras sig-nals originating from the membrane at different lipid raft-anchored Ras pools would have distinct effects on DNA methylation and demethylation [1,2,91,92], and histone 3 phosphorylation and acetylation machineries [55] Interestingly, this relationship between lipid rafts,

Fig 2 Epigenetic silencing of repair genes affects genetics O6-methylguanine DNA-methyltransferase (MGMT) gene silencing through promoter methylation demonstrates how the loss of MGMT expression results

in G to A transitions of the K-Ras oncogene (the most frequently mutated isoform of Ras), and of p53 [1,5,6,41,99] MGMT, a DNA repair protein, removes mutagenic and cytotoxic adducts from the O6 position of guanine [41,183] O6-methylguanine often mispairs with thymine during replication, and it results in conversion from a guanine– cytosine (GC) pair to an adenine–thymine (AT) pair if the adduct is not removed.

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Ras and epigenetic states might be bilateral as well

because many lipid raft component encoding genes are

known to be regulated by DNA methylation

[2,91,110]

FAS, FAS ligand, FAS-associated death

domain and lipid raft-mediated FAS

signaling

FAS-triggered apoptosis is another critical process

which is an effector of Ras signaling and is tightly

associated with epigenetic deregulation The 36 kDa

cell surface cytokine receptor, FAS (TNFRSF6⁄ FAS ⁄

APT1⁄ APO1 ⁄ CD95, OMIM 134637) contains a

16-amino acid signal sequence followed by a mature

protein of 319 amino acids that contains a solo

trans-membrane domain and two specialized functional

domains; a FAS death domain and a FAS ligand

(FASL) binding domain [111] FAS-associated death

domain (FADD, OMIM 602457) protein is the

univer-sal adaptor-protein for apoptosis This FADD

medi-ates signaling of all known death domain-containing

members of the tumor necrosis factor (TNF) receptor

superfamily [112] The FADD gene contains two exons

and spans  3.6 kb [113] Northern blot analysis

revealed that FADD was expressed as a 1.6-kb mRNA

in many fetal and adult tissues [114] The death

domain of FADD is 25–30% identical to those of

FAS and the TNF receptor, TNFR1 (OMIM 191190)

Natural ligands, cognate agonist antibodies and

inter-actions of FADD with FAS at their respective death

domains trigger apoptosis through FAS and TNFRI

High expression of FADD in mammalian cells induces

apoptosis, which can be blocked by Crma, a Pox-virus

gene product that also blocks FAS-induced apoptosis

[115,116]

The FAS protein shows structural homology with a

number of cell surface receptors, including TNFR1

and the low-affinity nerve growth factor receptor It

has been shown that following activation of T cells,

the FAS receptor is rapidly induced The interaction

between FAS and FASL induces cell death that occurs

in a cell-autonomous manner, similar to the classic

apoptotic sequence [117,118] FAS activates caspase 3

by inducing the cleavage of the caspase zymogen to its

active subunits and by stimulating the denitrosylation

of its active site thiol [119]

Myc-induced apoptosis requires interaction between

FAS and FASL on the cell surface [120] Hueber et al

established the dependence of Myc on FAS signaling

for its potent cell killing activity [120,121] The

path-way leading to apoptosis by FAS cross-linking with

FASL results in the formation of a death-inducing

signaling complex (DISC) composed of FAS, the signal adaptor protein FADD, and procaspase 8 and 10, and the caspase 8⁄ 10 regulator C-FLIP [91,122,123] Yeh

et al.[115] proposed that the interaction of FADD and FAS through their C-terminal death domains unmasks the N-terminal effector domain of FADD, allowing it

to recruit caspase 8 (CASP-8; 601763) to the FAS sig-naling complex This results in activation of a cysteine protease cascade, which leads to cell death Apoptosis triggered by infection, radiation or chemotherapeutic drugs is also mediated by FAS This process involves modification, placement in membrane, aggregation in lipid rafts and internalization of the FAS–DISC com-plex [91,124–130] Internalization of FAS with either lipid rafts or an endosomal compartment may deter-mine which signaling pathways are involved When internalization of FAS is blocked, the receptor cannot induce apoptosis and instead remains fully engaged, most probably in activating nonapoptotic⁄ proliferative pathways [112,115,125,127,131]

Inter-relation of genetic and epigenetic alterations in cancer

It is now evident, as discussed above, that changes occurring in cancer cells, including chromosomal instability, an increased propensity for mutation, acti-vation of oncogenes, silencing of tumor suppressor genes and inactivation of DNA repair systems are a result of both genetic and epigenetic abnormalities The correlation between the status of -CpG-island hypermethylation and⁄ or mutations in critical genes shows that, for virtually every tumor type, both gene-specific hypermethylation and distinct genetic altera-tions over time are major driving forces in neoplastic development But naturally occurring mutations of specific genes in somatic cells are infrequent, because under normal conditions maintenance of genomic integrity is guarded by a complex array of DNA monitoring and repair enzymes Karyotypic order is also guaranteed by other molecular guards, such as cell-cycle check-points that operate at critical times during mitotic division Together, these systems ensure that mutations are rare events, so rare indeed that the multiple mutations known to be present in tumor cells, which are necessary for cancer progres-sion, are low probability events within a normal human life span However, during oncogenesis epige-netic silencing of genes encoding DNA repair proteins (for example, MGMT) may cause retention of mutants as well as encourage neo-mutants [1–6,41] The FAS apoptotic pathway is one of the most promising targets of this process

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FAS mutations in cancer and other

diseases

Several lines of evidence have highlighted that perhaps

all tumor cells express FAS, but in many cases the

gene is mutated encoding a nonfunctional protein

Some cancers such as papillary thyroid carcinoma,

however, do express functional FAS [132] Analysis of

the entire FAS coding region in micro-dissected biopsy

samples from 21 burn scar-related squamous cell

carci-nomas revealed somatic point mutations in all of the

splice sites from three patients [133] The mutations

were located in all three domains of the protein: the

death domain, ligand-binding domain and

transmem-brane domain of the FAS gene Analyses of the other

FAS alleles in tumors carrying the N239D and C162R

mutations indicated loss of heterozygosity, and

expres-sion of FAS was confirmed in all tumors with FAS

mutations [91,133]

In contrast to the situation in burn scar-related

squamous cell carcinoma, no mutations were detected

in 50 cases of conventional squamous cell carcinoma

[133] This difference in mutation of the FAS gene is

interesting because burn scar-related squamous cell

carcinoma is usually more aggressive than

conven-tional squamous cell carcinoma It was therefore

sug-gested that somatic mutations in the FAS gene may

contribute to the development and⁄ or progression of

burn scar-related squamous cell carcinoma The

fol-lowing mutations in the gene encoding FAS were

iden-tified: a 957A-to-G transition resulting in an N239D

substitution in the FAS death domain; a 547A-to-G

transition resulting in an N102S substitution in the

FAS ligand-binding domain; a 726T-to-C transition

resulting in a C162R substitution in the FAS

trans-membrane domain [133] Zhang et al [134] genotyped

1000 Han Chinese lung cancer (211980) patients

and 1270 controls for two functional polymorphisms

in the promoter regions of the FAS and FASL

genes, -1377G-to-A (134637.0021) and -844T-to-C

(134638.0002), respectively Compared with

noncarri-ers, there was an increased risk of developing lung

cancer for carriers of either the FAS -1377AA or the

FASL -844CC genotype; carriers of both homozygous

genotypes had a more than fourfold increased risk

[134] Their results further support the concept that

the inactivation of FAS- and FASL-triggered

apopto-sis pathway plays an important role in human

carcino-genesis [91,135]

A heterozygous mutation in the FAS gene in five

unrelated children (134637.0001–134637.0005), with a

rare autoimmune lymphoproliferative (lpr) syndrome

was identified by Fisher et al [136] The disease is

characterized by massive nonmalignant lymphadenopa-thy, heightened autoimmunity and expanded popula-tions of TCR-CD3(+)CD4())CD8()) lymphocytes, resulting from defective FAS-mediated T-lymphocyte apoptosis While delineating the prognostic markers for the disorder, Sneller et al [137] further analyzed one of the patients studied by Fisher et al., and pointed out its resemblance to autosomal recessive lpr⁄ gld (generalized lymphoproliferative disorder) mouse The lpr and gld mice bear mutated genes for FAS and FASL, respectively The murine autosomal recessive lpr phenotype is characterized by lymphade-nopathy, hypergammaglobulinemia, multiple autoanti-bodies and the accumulation of large numbers of nonmalignant CD4, CD8 and T cells Affected mice usually develop a systemic lupus erythematosus-like autoimmune disease, and a defect in the negative selection of self-reactive T lymphocytes in the thymus The mouse lpr phenotype is identical to the phenotype displayed by human patients bearing mutated FAS [138,139]

Epigenetic downregulation of apoptosis – the role of the Ras signaling pathway

In addition to genetic mutations in FAS as discussed above, FAS is silenced by epigenetic mechanisms in several cancers Several lines of evidence suggest that the trigger for methylation of the FAS gene is activa-tion of the Ras fi Raf fi MEK fi ERK fi Elk signaling pathway [1,91,98] Methylation of the FAS gene is associated with loss of FAS expression in anti-gen-specific cytotoxic T cells [140] There is evidence for involvement of DNA methylation in silencing of FAS–FASL signaling and loss of apoptosis [141] Silencing of FASL and TRAIL-R1, TRAIL-R2 and Caspase-8 expression by DNA methylation has been linked to resistance of small cell lung cancer cells to FASL and TRAIL induced apoptosis [142] FAS pro-moter methylation in prostatic and bladder carcinomas and respective cell lines correlates with downregulation

of FAS expression [143]

H-Ras is linked to the silencing of FAS-triggered apoptosis through DNA methylation Peli et al [144] reported that oncogenic H-Ras downregulated FAS

by DNA methylation It was suggested that the phosphatidylinositol 3-kinase pathway was involved in mediating this effect of RAS The involvement of phosphatidylinositol 3-kinase points to the possibility that some of the known anti-apoptotic effects of PKB⁄ Akt kinase may be mediated, at least in part, by the downregulation of FAS expression through DNA

Trang 9

methylation [144] It remains to be seen whether this

effect of the Ras signaling pathway on DNA

methyla-tion is brought about by increase of DNMT1 as

pre-viously reported [102] or through activation of

factor(s) that recruits DNMT to specific targets such

as apoptosis

The epigenetic downregulation of apoptosis

path-ways involves additional genes in various types of

tumors In neuroblastomas and neurobalstoma cell

lines, which are resistant to apoptosis induced by

TRAIL, CASP-8 and the FLIP gene, and in tissues

adjacent to tumors the CASP-8 gene is

hypermethyla-ted [145] The FLIP protein is a negative regulator of

CASP-8, and the methylation of CASP-8 and FLIP

genes is somewhat correlated [91,125,145]

Mechanisms of silencing of FAS in

response to Ras activation

How does Ras activation cause methylation and

epige-netic silencing of FAS and other apoptosis-related

genes? Activated Ras epigenetically silences FAS

expression in mouse NIH3T3 cells [143], and in human

K-Ras transformed cell line, HEC1A [98]

Twenty-eight ‘Ras epigenetic silencing effector’ (RESE) genes

were discovered in a genome-wide functional screen

[98] Nine RESEs were found to be bound to different

regions of the FAS promoter in K-Ras-transformed

NIH3T3 cells [98], whereas in nontransfected NIH3T3

cells only one RESE (NPM2) was associated with the

FAS promoter It was therefore proposed that these

nine RESEs were recruited to specific regions of FAS

promoter in response to expression of oncogenic

K-Ras and are involved in the recruitment of DNMT1

and other chromatin modifiers to the promoter,

result-ing in DNA methylation and epigenetic silencresult-ing In

support of this hypothesis, knockdown of any of the

28 RESEs in K-Ras-transformed NIH3T3 cells

resulted in an absence of DNMT1 on the FAS

promoter, demethylation of the FAS promoter and

induction of FAS expression

What are the biochemical and cellular functions of

other RESEs? Among the 28 RESE proteins

discov-ered using a functional genomics approach there

are transcriptional activators and repressors (CTCF,

EID1, E2F1, RCOR2 and TRIM66⁄ TIF1D),

sequence-specific DNA-binding proteins (SOX14,

ZCCHC4 and ZFP345B), histone methyltransferases

(DOT1L, EZH2 and SMYD1), histone deacetylase

(HDAC9), histone chaperones (ASF1A and NPM2),

DNMT1 and several Polycomb group proteins (BMI1,

EED and EZH2) Several recent studies have linked

Polycomb proteins to abnormal DNA methylation and

gene silencing [1,146–148] It is surprising that one of the nuclear RESEs is BAZ2A⁄ TIP5, previously known

to be involved only in repression of RNA polymer-ase I-directed ribosomal gene transcription [149] A number of RESEs were substantially upregulated at the transcriptional or post-transcriptional level in K-Ras-transformed NIH3T3 cells compared with non-transformed NIH3T3 cells, explaining at least in part, how K-Ras activates this silencing pathway (Fig 3A) Treatment of K-Ras NIH3T3 cells with the demethy-lating drug 5-aza-CdR resulted in FAS re-expression, supporting the hypothesis that FAS is regulated by DNA methylation in Ras-transformed cells [98]

Epigenetic inactivation of the RAS effector homolog RASSF1

Genes encoding human RAS effector homolog, RASSF1 (OMIM, 605082) family proteins, along with several putative tumor-suppressor genes are located at chromosome 3p21 [83,150–152] RASSF1 produce eight transcripts, A–H, derived from alternative splic-ing and promoter usage [152–154] The RASSF1 gene contributes to the spatiotemporal regulation of mitosis through a number of regulatory mechanisms that cooperate to restrict the activity of APC⁄ C to a spe-cific period in the cell cycle [153–155] Mechanistic roles for RASSF1A in inducing apoptosis in cancer cells and solid tumors are emerging [156–158] RASSF1A function was missing in a variety of solid tumors and cancer cell lines, including small cell lung cancer and prostate [150–156] DNA methylation of the CpG island promoter sequence of RASSF1A was implicated in its silencing [16,155] RASSF1A is the most frequently methylated gene in both primary tumors and cell lines and in a group of nine genes mapped in 175 primary pediatric tumors and 23 tumor cell lines RASSF1A methylation was tumor specific and absent in adjacent nonmalignant tissues [157] RASSF1A gene silencing is also associated with aber-rant methylation and histone deacetylation in a variety

of other cancers [158–163] RASSF2 methylation and inactivation is a consequence as well of K-Ras-induced oncogenic transformation [164] Apart from Ras-regu-lated methylation of RASSF1A, Ras and RASSF1A have direct physical interaction in cellular physiology [155,160]

Lipid raft facilitated Ras signaling and chromatin modification

Clustering of raft-associated receptors, like epidermal growth factor receptor, facilitates the early step of

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