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Tiêu đề Mixed lineage leukemia: histone H3 lysine 4 methyltransferases from yeast to human
Tác giả Shivani Malik, Sukesh R. Bhaumik
Trường học Southern Illinois University School of Medicine
Chuyên ngành Biochemistry and Molecular Biology
Thể loại Minireview
Năm xuất bản 2010
Thành phố Carbondale
Định dạng
Số trang 17
Dung lượng 222,51 KB

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Nội dung

These complexes have a catalytic ATPase subunit with Keywords ASH1; ASH2; COMPASS; histone H3 lysine 4; histone methyltransferase; MLL; Set1; TAC1; TRX; WDR5 Correspondence S.. Abbreviat

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Mixed lineage leukemia: histone H3 lysine 4

methyltransferases from yeast to human

Shivani Malik and Sukesh R Bhaumik

Department of Biochemistry and Molecular Biology, Southern Illinois University School of Medicine, Carbondale, IL, USA

Introduction

The DNA in eukaryotes is compacted in the form of

chromatin The fundamental unit of chromatin is the

nucleosome which consists of a core histone particle

with 146 bp of DNA wrapped around it [1,2] The core

histone particle comprises a tetramer of histones H3 and

H4 and dimers of histones H2A and H2B [2] Each of

these histones has a structured core globular domain

and an unstructured flexible N-terminal tail protruding

from the core domain The linker histone H1 associates

with the core domain to form a higher order structure,

thus further compacting the DNA [3,4] Such

compac-tion of DNA in a higher order chromatin structure

makes it inaccessible for proteins involved in different

DNA-transacting processes such as transcription, repli-cation, recombination and DNA repair However, the chromatin structure has to be dynamic in nature in order for DNA-transacting processes to occur [5–10], and such dynamic states are regulated by ATP-depen-dent chromatin remodelers as well as by ATP-indepen-dent histone covalent modifications

There are several ATP-dependent chromatin remo-delers These include the switching–defective⁄ sucrose non-fermenting (SWI⁄ SNF), imitation switch (ISW1), nucleosome remodeling and histone deacetylation (Mi-2⁄ NuRD), and INO80 complexes [11–25] These complexes have a catalytic ATPase subunit with

Keywords

ASH1; ASH2; COMPASS; histone H3

lysine 4; histone methyltransferase; MLL;

Set1; TAC1; TRX; WDR5

Correspondence

S R Bhaumik, Department of Biochemistry

and Molecular Biology, Southern Illinois

University School of Medicine, Carbondale,

IL 62901, USA

Fax: +1 618 453 6440

Tel: +1 618 453 6479

E-mail: sbhaumik@siumed.edu

(Received 16 November 2009, revised

12 January 2010, accepted 22 January

2010)

doi:10.1111/j.1742-4658.2010.07607.x

The fourth lysine of histone H3 is post-translationally modified by a methyl group via the action of histone methyltransferase, and such a covalent modification is associated with transcriptionally active and⁄ or repressed chromatin states Thus, histone H3 lysine 4 methylation has a crucial role

in maintaining normal cellular functions In fact, misregulation of this covalent modification has been implicated in various types of cancer and other diseases Therefore, a large number of studies over recent years have been directed towards histone H3 lysine 4 methylation and the enzymes involved in this covalent modification in eukaryotes ranging from yeast to human These studies revealed a set of histone H3 lysine 4 methyltransfe-rases with important cellular functions in different eukaryotes, as discussed here

Abbreviations

ASH1, absent, small or homeotic discs 1; ASH2, absent, small or homeotic discs 2; BRM, brahma; CBP, CREB-binding protein; EcR, ecdysone receptor; HAT, histone acetyl transferase; H3K4, histone H3 lysine 4; HMT, histone methyltransferase; MLL, mixed lineage leukemia; MOF, male absent on the first; Paf1, RNA polymerase II-associated factor 1; PcG, polycomb group; PHD, plant homeodomain; TAC1, trithorax acetylation complex 1; TRR, trithorax-related; TRX, trithorax.

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DNA-dependent ATPase activity ATP-independent

histone covalent modifications are acetylation,

phos-phorylation, ubiquitylation, methylation, sumoylation

and ADP ribosylation [8,10,26–33] Although most of

these modifications occur on the N-terminal tails of

histones, some also occur on the C-terminal tails of histones H2A and H2B [29,30,34] and the core region

of histone H3 [30,31,35,36] These covalent modifica-tions have profound effects on chromatin structure and hence gene regulation [5,8,9,30,33]

H3

N-A R T K R K S T K R K K R K S K -C

79

me A

B

me

Set1 (Sc)

Set1 (Sp)

SET1 (Ce)

TRX (Dm)

ASH2 (Dm)

TRR (Dm)

ASH1 (Dm)

ASH1 (Hs)

MLL 1-4 (Hs)

SET1A (Hs)

SET1B (Hs)

SMYD3 (Hs)

SET7/9 (Hs)

Meisetz (Hs)

Clr4 (Sp) SU(VAR)3-9 (Dm) G9a (Dm) ASH1 (Dm) Eu-HMTase 1 (Hs) SUV39 H1 (Hs) SUV39 H2 (Hs) G9a (Hs) ESET or SETDB1 (Hs)

MES-2 (Ce)

E (Z) (Dm) G9a (Dm) EZH2 (Hs) EZH1 (Hs) G9a (Hs)

Set2 (Sc) Set2 (Sp) Set2 (Dm) ASH1 (Dm) NSD1 (Hs) SETD2/HYPB (Hs)

Dot1 (Sc) Dot1 (Sp) DOT1L (Hs)

Gene activation, telomeric silencing

& DNA repair

Hetero-chromatin formation

& silencing

Hetero-chromatin formation

& silencing

Inhibition

of intragenic transcription

& hetero-chromatin spreading, &

regulation of transcriptional elongation

Gene activation

& silencing, splicing,

& DNA repair

H4

N-S G R G K G G K G L G K G G A K R H R K V -C

me

Heterochromatin formation & silencing

Set9 (Sp) Pr-SET7 (Dm) ASH1 (Dm) SUV4-20 (Hs) Pr-SET7/SET8 (Hs)

Fig 1 Methylation of different lysine (K) residues of histones H3 (A) and H4 (B) with associated methylases and functions in genome expression and integrity Sc, Saccharomyces cerevisiae; Sp, Schizosaccharomyces pombe; Ce, Caenorhabditis elegans; Dm, Drosophila melanogaster; Hs, Homo sapiens.

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The lysine (K) residues of histones H3 and H4 can

be mono-, di- and trimethylated, and such methylation

is associated with active and⁄ or repressed chromatin

(Fig 1) Thus, histone methylation is linked to diverse

cellular regulatory functions [27,30,31,33] Indeed,

sev-eral studies have implicated histone methylation in

var-ious types of cancer and other diseases [30,33,37–39]

Therefore, a large number of studies over several years

have focused on histone methylation at different K

residues and the enzymes involved in this covalent

modification in diverse eukaryotes [27,30,31,33,40–45]

These studies have revealed several histone

meth-yltransferases (HMTs) involved in the K methylation

of histones H3 and H4 with crucial roles in

maintain-ing normal cellular functions in eukaryotes rangmaintain-ing

from yeast to humans (Fig 1) Here, we discuss

his-tone H3 lysine 4 (H3K4) methylation and the HMTs

involved in this covalent modification, highlighting the

similarities and differences in several eukaryotes such

as Saccharomyces cerevisiae (budding yeast),

Schizosac-charomyces pombe (fission yeast), Caenorhabditis

elegans (roundworm), Drosophila melanogaster (fruit

fly), Mus musculus (mouse) and Homo sapiens (human)

H3K4 methylation and HMTs in

Saccharomyces cerevisiae

In S cerevisiae, H3K4 methylation is involved in the

stimulation of transcription [29–31,33] Further, H3K4

methylation in S cerevisiae has been implicated in

silencing at telomeres, ribosomal DNA and the

mat-ing-type locus [30,33,46,47] Thus, H3K4 methylation

participates in both gene activation and repression

The enzyme responsible for this covalent modification

was first identified in a multiprotein complex, named

COMPASS, in S cerevisiae [48] COMPASS consists

of the catalytic subunit, Set1, and seven other proteins

(Cps60⁄ Bre2, Cps50 ⁄ Swd1, Cps40 ⁄ Spp1, Cps35 ⁄ Swd2,

Cps30⁄ Swd3, Cps25⁄ Sdc1 and Cps15⁄ Shg1)

(Tables 1–3) [29,33,48,49] Set1 is essential for mono-,

di- and trimethylation of histone H3 at K4 [29,30,33, 48,49] Set1 is enzymatically active only when assem-bled into the multi-subunit COMPASS complex The ability of COMPASS to mono-, di- and trimethylate K4 of histone H3 depends on its subunit composition For example, COMPASS lacking Cps60⁄ Bre2 cannot trimethylate K4 of histone H3, whereas the Cps25 subunit of COMPASS is essential for histone H3 K4 di- and trimethylation [29,33,49,50] The COMPASS complex preferentially associates with RNA polymer-ase II which is phosphorylated at Ser 5 in its C-termi-nal domain at the onset of transcriptioC-termi-nal elongation [33,49,51–54] The interaction between COMPASS and RNA polymerase II is further facilitated by the RNA polymerase II-associated factor 1 (Paf1) complex which associates with the coding sequence in an RNA polymerase II-dependent manner during transcriptional elongation [33,49,51–54] Thus, COMPASS is found to

be predominantly associated with the coding sequences

of active genes [33,49,51,54,55], and the coding sequences of the actively transcribing genes are there-fore trimethylated at the K4 of histone H3 [33,51,54– 58]

Interestingly, the methyltransferase activity of the COMPASS complex is intimately regulated by ubiqui-tylation of histone H2B at K123 [30,33,59–63] Both di- and trimethylation of histone H3 at K4 are impaired in the absence of histone H2B K123 ubiqui-tylation, which is catalyzed by E2 ubiquitin conjugase and E3 ubiquitin ligase, Rad26 and Bre1, respectively However, histone H2B K123 ubiquitylation does not regulate H3K4 monomethylation [33,62,64] Such a trans-tail cross-talk between histone H2B K123 ubiquitylation and H3K4 di- and trimethylation is mediated via alteration of the subunit composition of COMPASS [33,55] It was recently demonstrated that histone H2B K123 ubiquitylation is essential for the recruitment of Cps35⁄ Swd2 independent of Set1 [33,55] Set1 maintains the structural integrity of the COMPASS complex [33,55] COMPASS without

Table 1 The histone H3 lysine 4 methyltransferases in different eukaryotes (references are cited in the text).

Saccharomyces cerevisiae Schizosaccharomyces pombe Caenorhabditis elegans Drosophila Mammals (mouse and human)

ASH1 ASH2 TRR

MLL1 MLL2 MLL3 MLL4 SET1A SET1B SET7 ⁄ 9 SMYD3 ASH1 Meisetz

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Cps35⁄ Swd2 is consistently recruited to the coding

sequence of the active gene in an RNA polymerase

II-dependent manner in the absence of histone H2B

K123 ubiquitylation [33,55] COMPASS without

Cps35⁄ Swd2 monomethylates K4 of histone H3, but

does not have catalytic activity for di- and

trimethyla-tion of histone H3 at K4 [33,55] When histone H2B is

ubiquitylated by the combined actions of Rad26 and

Bre1, it recruits Cps35⁄ Swd2, which interacts with the

rest of COMPASS recruited by elongating RNA

poly-merase II Such interaction leads to the formation of a

fully active COMPASS capable of H3K4 mono-,

di-and trimethylation [33,55] Thus, H3K4 methylation is

regulated by upstream factors involved in histone H2B

K123 ubiquitylation Further, H3K4 methylation is controlled by a demethylase with the Jumonji C (JmjC) domain, namely Jhd2, which specifically deme-thylates the trimethylated K4 of histone H3 (Table 4) [65] Such demethylation provides an additional level

of regulation of H3K4 methylation in S cerevisae

H3K4 methylation and HMTs in Schizosaccharomyces pombe

Although the first H3K4 methyltransferase was identified in S cerevisiae, the chromatin structure in

S cerevisiae is not similar to that of Sch pombe or higher eukaryotes For example, S cerevisiae lacks

Table 2 The components of characterized histone H3 lysine 4 methyltransferase complexes in different eukaryotes (references are cited in the text).

Saccharomyces cerevisae Schizosaccharomyces pombe Caenorhabditis elegans Drosophila Mammals COMPASS ⁄ Set1C

Set1

Cps60 ⁄ Bre2

Cps50 ⁄ Swd1

Cps40 ⁄ Spp1

Cps35 ⁄ Swd2

Cps30 ⁄ Swd3

Cps25 ⁄ Sdc1

Cps15 ⁄ Shg1

Set1C Set1 Ash2 Swd1 Spp1 Swd2 Swd3 Sdc1 Shg1

COMPASS-like complex SET-2 ⁄ SET1

ASH2 ⁄ Y17G7B.2 CFPL-1 SWD-3 SWD-2 ⁄ C33H5.6 SPP-1 ⁄ F52B11.1 DPY-30

TAC1 TRX CBP SBF1 ASH1 ASH1

? ASH2 ASH2

? TRR TRR

?

MLL1 MLL1 ASH2L WDR5 RBBP5 DPY-30 HCF1 ⁄ HCF2 Menin MOF MLL2 MLL1 ASH2L WDR5 RBBP5 DPY-30 Menin HCF2 RPB2 MLL3 ⁄ MLL4 MLL3 ⁄ MLL4 ASH2L WDR5 RBBP5 DPY-30 NCOA6 PA1 PTIP UTX SET1A ⁄ SET1B SET1A ⁄ SET1B ASH2L WDR5 RBBP5 WDR82 ⁄ SWD2 a

CFP1 ⁄ CGBP a

DPY-30 HCF1

a Wdr82 and CFP1 ⁄ CGBP are present in human SET1A and SET1B complexes, but not in mouse [117,165,166].

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homologs of the repressive histone H3K9

methyltransfe-rases and the heterochromatin proteins (e.g HP1)

present in Sch pombe or higher eukaryotes [66–68]

Thus, Sch pombe serves as a better eukaryotic model

system to study the roles of H3K4 methylation in

regulation of chromatin structure and gene expression

As in S cerevisae, H3K4 methylation in Sch pombe is

catalyzed by a SET domain-containing protein, Set1

The Sch pombe Set1 protein is homologous to S

cerevi-siae Set1 Set1 proteins in budding and fission yeasts

share a high degree of similarity in their SET domains

However, these two proteins exhibit 26% sequence

iden-tity overall [69] The N-termini of Set1 in S cerevisae

and Sch pombe are considerably different [69–71] Such

difference might have crucial roles in governing the

specific functions of Set1 in S cerevisae and Sch pombe

For example, a recombinant Sch pombe Set1 methylates

K4 in a 20-amino acid peptide corresponding to the

N-terminal tail of histone H3 in vitro By contrast,

recombinant S cerevisae Set1 does not have

methyl-transferase activity in vitro Further, phylogenetic

analy-sis indicates that Sch pombe Set1 is more closely related

to human Set1 than to S cerevisae Set1 [69] Sch pombe

Set1 mutants have slow growth, exhibit

temperature-sensitive growth defects and have a slightly longer

dou-bling time compared with wild-type cells [69]

DNA sequence analysis reveals that homologs of the components of S cerevisae COMPASS are also pres-ent in Sch pombe Indeed, Set1 methyltransferase com-plex (Set1C) has been purified in Sch pombe, which shares many features of S cerevisae COMPASS (Tables 1–3) However, these two complexes differ in several ways For example, the Ash2 component of Set1C in Sch pombe has a plant homeodomain (PHD) finger domain, whereas the homologous protein, Cps60⁄ Bre2 (Table 3), in S cerevisae does not [70] The Cps40⁄ Spp1 component (that bears the PHD fin-ger domain) is required for methylation in Sch pombe, but not in S cerevisae [70] Furthermore, Set1C in Sch pombeshows a hyperlink to Lid2C (little imaginal discs 2 complex) through Ash2 and Sdc1 [70] How-ever, such a hyperlink is absent in S cerevisae In addition, the identified hyperlink, Swd2 (which is also

a subunit of the cleavage and polyadenylation factor)

in S cerevisae COMPASS is not found in Sch pombe [70–72] Together, these observations support the fact that the Set1 HMTs from S cerevisae and Sch pombe are highly conserved (Tables 1–3), but their proteomic environments appear to differ However, such differ-ences in the proteomic environments may be related to the absence of histone H3 K9 methylation in S cerevi-siae, as suggested previously [70]

Table 4 Histone H3 lysine 4 demethylases in different eukaryotes (references are cited in the text) me 3 , trimethyl; me 2 , dimethyl; me 3 ⁄ 2 , tri- and dimethyl; and me2⁄ 1, di- and monomethyl.

Saccharomyces cerevisae Schizosaccharomyces pombe Caenorhabditis elegans Drosophila Mammals (mouse and human)

Swm2 (me 2 )

SPR-5 (me 2 ) T08D10.2 (me 2 ) R13G10.2 (me2)

Lid (me 3 ) LSD1 (me 2 ⁄ 1 )

JARID1A (me 3 ⁄ 2 ) JARID1B (me3⁄ 2) JARID1C (me3⁄ 2 ) JARID1D (me 3⁄ 2 )

Table 3 Homologous subunits of histone H3 lysine 4 methyltransferase complexes in different eukaryotes (references are cited in the text) Saccharomyces

cerevisiae

Schizosaccharomyces pombe

Caenorhabditis

TRR

Menin; HCF1 ⁄ HCF2; NCOA6; PA1;

PTIP; UTX; MOF; SET7 ⁄ 9; SMYD3; Meisetz

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H3K4 methylation is correlated with active

chroma-tin in Sch pombe [69], as in S cerevisae However,

unlike in S cerevisiae, H3K4 methylation is not

required for silencing and heterochromatin assembly at

the centromeres and mating type locus in Sch pombe

[69], possibly because of the presence of repressive

histone H3 K9 methylation in Sch pombe [69]

Fur-thermore, previous studies [69] have demonstrated that

H3K4 methylation is correlated with histone H3

acety-lation in Sch pombe, and hence is associated with

active genes However, transcriptional stimulation by

H3K4 methylation is also closely regulated by histone

demethylase, LSD1, which is absent in S cerevisae

LSD1 is an amine oxidase which demethylates the

K residue in a FAD-dependent manner Because it

functions through oxidation, it can only demethylate

mono- and dimethylated K4 of histone H3 There are

two LSD1-like proteins, namely Swm1 and Swm2

(after SWIRM1 and SWIRM2), in Sch pombe

(Table 4) [73,74] which form a complex and are

involved in the demethylation of methylated K4 and

K9 of histone H3 Such a demethylation process has

important roles in the regulation of chromatin

struc-ture and hence gene expression

H3K4 methylation and HMTs in

Caenorhabditis elegans

C elegansis a multicellular, yet simple, eukaryotic

sys-tem with technical advantages for studying the

chro-matin structure in greater detail Thus, C elegans can

serve as a model system to understand the role of

histone covalent modifications in developmental

pro-cesses As in S cerevisae and Sch pombe, H3K4

meth-ylation has an important role in promoting

transcription in C elegans [75] However, early C

ele-gans embryos have a transcriptionally repressed

chro-matin state, even though both di- and trimethylation

of histone H3 at K4 are present in the chromatin of

the germline blastomere [75] Such repression perhaps

results from the lack of Ser 2 phosphorylation in the

C-terminal domain of the largest RNA polymerase II

subunit in the germline cells [76] Following division of

the germline lineage P4 cells into the primordial germ

cells, H3K4 methylation is lost [75] However, H3K4

methylation is regained prior to postembryonic

prolif-eration Such covalent modification activates gene

expression in the postembryonic germ cells [75,77]

The enzyme involved in H3K4 methylation in C

ele-ganswas identified recently via an RNAi screen of the

suppressors of heterochromatin protein mutants (hpl-1

and hpl-2) The RNAi screen identified set-2 as the

homolog of yeast SET1 [78] Further, several studies

have revealed that 2 (also known as

SET-2⁄ SET1) forms a complex with SWD-3, CFPL-1, DPY-30, Y17G7B.2⁄ ASH-2, C33H5.6 ⁄ WDR82 ⁄

SWD-2 and F5SWD-2B11.1⁄ SPP-1 (Tables 1 and 2) [78] These proteins are homologous to the budding yeast COM-PASS (Table 3) Thus, as in S cerevisae, SET-2⁄ SET1

in C elegans forms a COMPASS-like complex Fur-thermore, like in fission yeast, SET-2⁄ SET1 may be hyperlinked to the complex that is homologus to Sch pombe’s Lid2C-containing Sdc1 In support of this notion, DPY-30 in C elegans was found to be the homolog of fission yeast Sdc1 (Table 3), and it plays

an important role in dosage compensation [79] Thus,

it is likely that SET-2⁄ SET1 in C elegans is connected

to another complex via DPY-30, which remains to be elucidated

As in yeast, the subunits of the COMPASS-like complex in C elegans differentially regulate global H3K4 methylation [78] For example, no decrease in the global level of H3K4 methylation was observed upon RNAi-based depletion of swd-2 [78] A moderate decrease in H3K4 methylation was observed in the absence of Y17G7B.2⁄ ASH-2 Depletion of set-2, swd-3, cfpl-1 and dpy-30 led to a drastic decrease in global H3K4 methylation, with the most severe defect observed in swd-3 mutants However, unlike in yeast, the residual level of global H3K4 methylation was observed in the absence of SET-2⁄ SET1 activity [78] This observation suggests that additional H3K4 methyltransferase(s) may exist in C elegans

Although H3K4 methylation is associated with active transcription in C elegans, it is reset during gametogenesis A demethylase, SPR-5, has recently been identified in C elgans, and it shows 45% similar-ity with human LSD1 [80] SPR-5 is responsible for demethylation of dimethylated K4 of histone H3 (Table 4) It interacts with the (co)repressor for ele-ment-1-silencing transcription factor repressor protein, SPR-1 [81–83] Such an interaction is correlated with the repressive role of SPR-5 in gene regulation Like SPR-5, two other proteins in C elegans, namely T08D10.2 and R13G10.2, have a LSD1-like amine oxi-dase domain, as revealed by the NCBI Conserved Domain Search Program (Table 4) [84] Knockdown

of T08D10.2 by RNAi extends the longevity, thus implicating the role of histone methyaltion in regula-tion of aging [85]

Although studies in C elegans have been quite help-ful in understanding the role of H3K4 methylation in gene expression and development, the pattern of cell lineage in C elegans is highly invariant [86] However, the development of embryos of Drosophila and mam-mals largely relies on cellular cues, thus making it a

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more complex process Therefore, studies in Drosophila

will provide a better understanding of the regulatory

roles of H3K4 methylation in gene expression and

development

H3K4 methylation and HMTs in

Drosophila

Drosophilahas long been a model organism for studying

developmental processes, because development in

humans and Drosophila are homologous processes

Dro-sophilaand humans share a number of related

develop-mental genes working in conserved pathways Studies

analyzing the interplay of the SET domain-containing

trithorax group (trxG) and polycomb group (PcG)

pro-teins in regulating transcription patterns during

develop-ment and differentiation have been an area of extensive

research in Drosophila [87–91] PcG and trxG proteins

play a crucial role in the epigenetic control of a large

number of developmental genes, including the Hox

(Homeotic) genes [92–99] Hox are a cluster of genes

that defines the anterior–posterior axis and segment

identity during early embryogenesis The expression

pat-tern of the Hox genes is established early in development

and is propagated in appropriate cell lineages [100]

However, transcription of Hox genes is closely regulated

by the antagonistic actions of PcG and trxG proteins

through different patterns of histone methylation The

PcG protein, E(Z), methylates histone H3 at K27 [100]

Further, histone H3 methylated at K27 is recognized by

a chromodomain-containing PcG protein, PC [100]

These events repress Hox and other target genes

How-ever, the trxG proteins such as trithorax (TRX) and

absent, small or homeotic discs 1 (ASH1) are H3K4

methyltransferases which promote transcription of the

target genes including Hox [100] Thus, a close interplay

between the specific PcG and trxG proteins maintains a

tight regulation of Hox gene expression Consistently,

genetic studies in Drosophila have revealed that

muta-tions in specific PcG and trxG genes result in flies with

homeotic transformations because of the misregulation

of Hox genes [100–103]

As mentioned above, TRX is a member of the trxG

proteins with H3K4 methyltransferase activity in

Drosophila, and it contains SET and PHD finger

domains TRX is the homolog of yeast Set1 in

Dro-sophila, and is an integral component of a 1 MDa

complex, called trithorax acetylation complex 1

(TAC1) TAC1 consists of TRX, CREB-binding

pro-tein (CBP) and an anti-phosphatase SBF1 (Tables 2

and 3) [104] Like mammalian CBP, Drosophila CBP

has histone acetyl transferase (HAT) activity [104]

Thus, TAC1 possesses both HMT and HAT activities

[94,104] which are associated with active transcription The components of TAC1 are found to be associated with specific sites on salivary gland polytene chromo-somes, including Hox genes [104], and thus exist together in vivo Mutations in either trx or the gene encoding CBP reduce the expression of a Hox gene, namely Ultrabithorax (Ubx) [104] Thus, the two differ-ent enzymatic activities of TAC1 are closely linked to Hox gene expression [104] Moreover, the HAT activ-ity of TAC1 may be counteracted by the deacetylase activity of the PcG complex, ESC⁄ E(Z), accounting in part for the antagonistic functions of the trxG and PcG protein complexes on chromatin Unlike its role

in the regulation of Hox gene expression, TAC1 also promotes transcription of heat shock genes in a differ-ent mechanism through activation of poised or stalled RNA polymerase II Heat shock genes are rapidly expressed by heat shock factor and other transcription factors [94,105] TAC1 is recruited to several heat shock gene loci following heat induction, and conse-quently, its components are required for heat shock gene expression [94,105] Smith et al [94] demonstrate that TAC1 associates with transcription-competent stalled RNA polymerase II at the heat shock gene, and subsequently modifies histone H3 by methylation and acetylation Such modifications of histone H3 facilitate stalled RNA polymerase II to begin transcriptional elongation [94,105] In contrast to the results at heat shock genes, poised or stalled RNA polymerase II is not found at the Hox genes [94,105] Thus, TAC1 appears to regulate the transcription of Hox and heat shock genes in distinct pathways

In addition to TRX, two other trxG proteins, namely ASH1 and ASH2, methylate K4 of histone H3 [106–110] Mutations in the Ash1 and Ash2 genes gen-erate abnormal imaginal discs in flies [106–110], consis-tent with their roles in the regulation of Hox gene expression Amorphic and antimorphic mutations in the Ash1 gene lead to a drastic decrease in the global level of H3K4 methyaltion [106–110] The catalytic domain of ASH1 is 588 amino acids long, and com-prises the SET domain and cysteine-rich pre-SET and post-SET domains [106,107] However, biochemical studies demonstrate that the 149-amino acid SET domain alone can methylate histone H3 at K4 in vitro [107] ASH1 also contains a PHD finger, and a associated homology domain [111] The bromo-associated homology domain of ASH1 might be responsible for protein–protein interactions during chromatin remodeling at the target genes ASH1 is an integral component of a large 2 MDa complex [112]

In addition to H3K4 methylation, the ASH1 complex also methylates K9 of histone H3 and K20 of

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histone H4 [106,107] Recently, Tanaka et al [113]

implicated ASH1 in methylation of histone H3 at K36

Apart from its role in histone methylation, ASH1 is

also linked to histone acetylation via its interaction

with CBP [114] which is an integral component of

TAC1 Thus, ASH1 and TAC1 appear to have

com-mon roles via CBP

Like ASH1, ASH2 is present in a 500 kDa complex

[112] ASH2 has been proposed to be the associated

form of Bre2 and Spp1 of S cerevisae COMPASS

[48,71,115] In mammals, ASH2 is a shared component

of different complexes including a HMT bound by

host cell factor 1 (HCF-1), Menin-containing complex

and the COMPASS counterpart [116–120], indicating

that it might be involved in the regulation of many

dif-ferent processes However, its role in histone

methyla-tion is not known Recently, Steward et al [121]

demonstrated that ASH2 in mammalian system has an

important role in H3K4 trimethylation Consistent

with this observation, Beltran et al [122] observed a

severe reduction in H3K4 trimethylation in ash2

mutants This observation indicates that ASH2 might

play a crucial role in H3K4 methyltransferase activity

However, ASH2 does not contain a SET domain, but

it has the PHD finger and SPRY domains [123] In

addition to its role in H3K4 methylation, ASH2 is also

linked to histone deacetylation through its interaction

with Sin3A, a histone deacetylase [116] Further,

ASH2 has been implicated in the regulation of

cell-cycle progression via its interaction with HCF-1 [116]

Like trxG proteins, a trithorax-related (TRR) protein

in Drosophila is also involved in the methylation of

his-tone H3 at K4 [124] TRR contains the SET domain,

and has H3K4 methyltransferase activity [124] TRR

functions upstream of hedgehog (hh) in the progression

of the morphogenic furrow [124] It also participates in

retinal differentiation [124] TRR and trimethylated

his-tone H3 at K4 are detected at the ecdysone-inducible

promoters of hh and BR-C (broad complex) [124]

Ecdysone functions through binding to a nuclear

recep-tor, ecdysone receptor (EcR), which heterodimerizes

with the retinoid X receptor homolog ultraspiracle

The heterodimer is then recruited to the promoters of

the target genes to regulate their expression, and hence

ecdysone triggers molting and metamorphosis Thus,

the association of EcR along with TRR and H3K4

methylation is also observed at the hh and BR-C

promoters following ecdysone treatment in cultured

cells [124] Consistent with these observations, H3K4

methylation is decreased at these promoters in embryos

lacking functional TRR [124] Thus, TRR appears to

function as a coactivator at the ecdysone-responsive

promoters by modulating the chromatin structure

H3K4 methylation functions as a platform for the binding of different chromatin remodelers One such remodeler is the BRM complex which contains at least seven proteins [112] Three components of the BRM complex are trxG proteins These are BRM (brahma), Osa and Moira However, these trxG protein compo-nents of the BRM complex do not have the SET domain as well as HMT activity The BRM complex is the homolog of the yeast SWI⁄ SNF complex, and shares four components including the ATPase BRM [112] BRM also contains a high-mobility-group B pro-tein, namely BAP111, which binds nonspecifically to the minor groove of the double-helix and bends the DNA [125,126] The BRM complex has ATP-depen-dent chromatin-remodeling activity Mutations in ash1 enhance brm mutations, suggesting that they might be functioning together [110] Consistent with this obser-vation, Beisel et al [106] demonstrated that epigenetic activation of Ubx transcription coincides with H3K4 trimethylation by ASH1 and recruitment of the BRM complex Similarly, mutations of ash2 and brm cause developmental defects in adult sensory organs includ-ing campaniform sensilla and mechanosensory bristles [108,127] Thus, although ASH1, ASH2 and BRM are the components of three distinct complexes, they appear to function in concert to regulate transcription Furthermore, the H3K4 methyltransferase activity of TAC1 has been implicated to be linked to the BRM complex [112,128] Such linkage is mediated by the interaction of TRX of TAC1 with the SNR1 compo-nent of the BRM complex [128] Together, these results indicate that H3K4 methylation and ATP-dependent chromatin remodeler, BRM, function in a concerted manner to regulate transcription Apart from the BRM complex, two other chromatin remo-delers, namely nucleosome remodeling factor and ATP-utilizing chromatin assembly and remodeling fac-tor, have been implicated in transcriptional stimulation through their binding to methylated-K4 of histone H3 which is mediated by TRX or other HMTs [100] Both nucleosome remodeling factor and ATP-utilizing chro-matin assembly and remodeling factor are ATP-depen-dent remodelers and carry ISW1 as an ATPase subunit [100] Thus, the HMTs mark a modification pattern on histone H3 at K4 that is ‘read’ by chromatin remodel-ers which, in turn, regulate the chromatin structure and hence gene expression

Apart from H3K4 methyltransferase and ATP-dependent chromatin remodeling activities, trxG pro-tein is also involved in histone demethylation Recent studies [129,130] have demonstrated that a trxG pro-tein, namely Lid, contains a JmjC domain and other functional domains found in mammalian Jumonji,

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AT-rich interactive domain 1 (JARID1) proteins Lid

has demethylase activity which can demethylate the

trimethylated form of histone H3 at K4 (Table 4)

Such demethylase activity of the trxG protein adds an

additional layer to gene regulation by the PcG and

trxG proteins Further, Lid interacts with the proteins

associated with heterochromatin formation such as

H3 K9 methyltransferase [Su(var)3-9], heterochromatin

protein (HP1) and deacetylase (RPD3) Thus, Lid

plays crucial roles in removing the activation marks,

hence facilitating gene silencing

The role of H3K4 methylation and its regulation in

Drosophilais largely conserved in mammals However,

the complexity of mammals demands a more intricate

mechanism of regulation in determining the cell

lin-eages and developmental fates Thus, a large number

of studies have focused on H3K4 methylation and

HMTs, and their roles in gene regulation with

implica-tions for development in mammals Below we discuss

H3K4 methyltransferases and H3K4 methylation in

mouse and humans with their regulatory roles in gene

expression

H3K4 methylation and HMTs in mouse

and humans

Histones are among the most conserved proteins

dur-ing evolution of eukaryotes As discussed for other

eukaryotes, roles of histone methylation in gene

regu-lation and development are largely conserved in

mam-mals Genomic studies have revealed that both mouse

and humans have  30 000 genes, and mouse has

orthologs for 99% of human genes (Mouse Genome

Sequencing Consortium, 2002) Given the close

conser-vation between these two systems, we have reviewed

progresses made towards H3K4 methylation and the

corresponding HMTs in both mouse and humans

H3K4 trimethylation patterns in mammals are similar

to yeast, and are associated with transcriptional start

sites H3K4 dimethylation, however, has a distinct

dis-tribution pattern Genomic mapping studies have

revealed that H3K4 dimethylation overlaps with H3K4

trimethylation in the vicinity of active genes [131,132]

However, significant H3K4 methylation is also

observed at the inactive genes within b-globin locus

This observation suggests that H3K4 methylation may

have important roles in maintaining the transcriptional

‘poised state’ [131], in addition to its role in

transcrip-tional stimulation

Although H3K4 methylation is usually localized to

punctuate sites, a unique pattern of H3K4 methylation

is found at the HOX gene cluster in mammals Broad

regions of continuous H3K4 methylation spanning

multiple genes with the intergenic regions are observed

at HOX gene clusters in mouse and humans [132] Unlike in yeast, which has just one H3K4 methyltrans-ferase, mammals have at least 10 (Table 1) [33,133– 138] Of these, six members containing the SET domain (MLL1, MLL2, MLL3, MLL4, SET1A and SET1B) belong to the mixed lineage leukemia (MLL) family bearing homology to yeast Set1 and Drosophila TRX (Tables 2 and 3) [30,33,49,133,134] Other H3K4 methyltransferases identified are ASH1, SET7⁄ 9, SMYD3 and a meiosis-specific factor, Meisetz [33,133– 138] The presence of several HMTs in humans might

be because of the role of different HMTs at different developmental stages in determining cell fate The MLL family of HMT proteins has an important role

in regulating HOX gene expression This is particularly significant because deregulation of HOX genes is asso-ciated with leukemia via rearrangements in the MLL1 gene In addition to HOX genes, MLL1 also targets non-HOX genes like p27 and p18 [139] Interestingly, deletions or truncations in MLL1, MLL2 and MLL3 have different phenotypes in mice [140–144] For example, deletion of MLL1 shows misregulation in a number of HOXA genes, including HOXA1 [140–143], whereas MLL2 controls expression of HOXB2 and HOXB5, and loss of MLL3 causes severe growth retar-dation and widespread apoptosis [141–143] Thus, MLL1, MLL2 and MLL3 appear to have nonredun-dant functions

As is true for yeast and other eukaryotes, MLL fam-ily HMTs are assembled into multi-subunit complexes (Table 2) These complexes have three common subun-its, WD repeat domain 5 (WDR5), retinoblastoma binding protein 5 (RBPB5) and Drosophila ASH2-like (ASH2L) [49,116,117,119,120,133,143] which form the core of the complex MLL SET is active only when it associates with the core complex, a feature reminiscent

of S cerevisae COMPASS [145] The WDR5 subunit

of the MLL complex is essential for binding of MLL HMT to dimethylated-K4 of histone H3 It is also a key player in the conversion of di- to trimethylation of histone H3 at K4 Consistent with this observation, a reduced level of H3K4 trimethylation is observed following knockdown of WDR5 Consequently, HOX gene expression in human cells is decreased signifi-cantly in the absence of WDR5 [146] Thus, WDR5 appears to play a crucial role in regulating the actitivi-ties of MLL HMTs

In addition to its role in H3K4 methylation, MLL complex interacts with a TATA-box binding protein (TBP)-associated factor component of transcription factor IID, components of E2F transcription factor 6 (E2F6) subcomplex, and MOF (a MYST family

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histone acetyl transferase involved in histone H3 K16

acetylation) [147] The interaction between the MLL1

complex and MOF is particularly interesting as both

histone H3 K4 methylation and histone H4 K16

acety-lation are marks of active transcription, and hint

towards the coordinated actions of histone H4 K16

acetylation and H3K4 methylation in transcriptional

regulation Indeed, it has been shown that MLL1

HMT and male absent on the first (MOF) HAT

acti-vities are required for the proper expression of HoxA9

[145,147] Further, the MLL3⁄ MLL4 complex

coor-dinates H3K4 methylation with demethylation of

histone H3 at K27 through its UTX subunit

[49,133,134,148,149] Furthermore, H3K4 methylation

is coupled to histone deacetylation by the interaction

of HMTs SET1⁄ ASH2 with SIN3 deacetylase [116]

Thus, HMTs in human appears to play diverse

func-tions in regulation of gene expression, and hence

development

In addition to its role in mammalian development,

H3K4 methylation also participates in the maintenance

of pluripotency It has been demonstrated that

embry-onic stem cells maintain ‘bivalent domains’ of

repres-sive (histone H3 K27 methylation) and activating

(H3K4 methylation) marks The bivalent domains

silence developmental genes in embryonic stem cells

while still preserving their ability to be activated in

response to appropriate developmental cues [150]

However, bivalent domains are also maintained at

other genes in fully differentiated cell types [151,152]

Methylation is dynamically regulated by

demethylas-es like LSD1 and JmjC domain-containing enzymdemethylas-es

Demethylase, LSD1, can demethylate mono- and

dimethylated-K4 of histone H3 (Table 4) LSD1 has

been shown to interact with repressors like

(co)repres-sor for element-1-silencing transcription factor and

activation complexes like MLL1 These observations

implicated that MLL1 is involved in transcriptional

activation as well as repression [33,49,153,154]

Deme-thylation of trimethylated-K4 of histone H3 is

cata-lyzed by JmjC domain-containing proteins Mammals

have four JARID family members with the JmjC

domain (Table 4) These are JARID1A or Rbp2,

JARID1B or PLU-1, JARID1C or SMCX and

JAR-ID1D or SMCY [33,155] Both H3K4

methyltransfe-rases and demethylases function in a coordinated

fashion to delicately regulate H3K4 methylation, and

hence gene expression

The significance of understanding regulation of

H3K4 methylation is exemplified by the occurrence

of cancers and other diseases following mutations of

H3K4 methyltransferases and demethylases or their

altered expression [33] For example, MLL1 is

translo-cated in leukemia; SMYD3 is overexpressed in colorec-tal cancer, liver, breast and cervical cancers; and the demethylase, LSD1, is overexpressed in prostate can-cer The implication of HMTs and demethylases in cancer and human diseases has led to the idea of utiliz-ing them as therapeutic targets In fact, biguanide and bisguanidine polyamine analogs inhibit the demethy-lase, LSD1, in human colon carcinoma cells Inhibition

of LSD1 facilitates expression of the aberrantly silenced genes in cancer cells [156] In addition to tar-geting a single enzyme, epigenetic therapy combining related proteins⁄ pathways is a promising alternative Thus, combinatorial therapy targeting HMTs, demeth-ylases, histone deacetdemeth-ylases, DNA methyltransferases and others may work efficiently in treating diseases caused by epigenetic misregulations In fact, a combi-natorial therapy targeting histone deacetylases and DNA methyltransferases has been shown to have a synergistic role in gene regulation, and clinical trials have consistently yielded encouraging results [157–161] Structural and functional studies have shown specific similarities and differences among the HMTs in diverse organisms In mammals, the core components of the MLL complex, RBBP5, WDR5 and ASH2L, form a structural platform with which the catalytic SET1 can associate, whereas Set1 in S cerevisiae is required for the integrity of the COMPASS complex In S

cerevisi-ae, Cps60⁄ Bre2 does not interact directly with Cps50⁄ Swd1 However, in mammals their homologs RBBP5 and ASH2L interact strongly in the complex These different interactions imply diverse regulatory mechanisms for the HMTs This suggests that in higher eukaryotes, the core complex can be similar, but different subunits can associate with this core com-plex at different stages of cell development to provide HMT activity Thus, there are several HMT complexes

in higher eukaryotes dedicated to diverse cellular roles Further, the C-terminal SET domain is invariant in different HMTs, although the N-terminal domains are divergent This enables the HMTs to associate with a broad spectrum of proteins to ensure the downstream events Given the increasing complexity in higher eukaryotes, the diversity of H3K4 HMTs is not sur-prising However, the conservation of the fundamental SET domain in these HMTs is intriguing In addition, many of the H3K4 HMTs share several domains⁄ com-ponents, indicating a common mechanism of action

Concluding remarks

Studies in several eukaryotes demonstrate the conser-vation of H3K4 HMTs from yeast to humans The roles of H3K4 methylation in gene regulation,

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