Berek Novak's Gyn 2019. Chapter 6 Molecular Biology and Genetics

CHAPTER 6

Molecular Biology and Genetics

KEY POINTS

1 The regulation and maintenance of normal tissue requires a balance between cell

proliferation and programmed cell death, or apoptosis.

2 Among the genes that participate in the control of cell growth and function, protooncogenes and tumor suppressor genes are particularly important.

3 Growth factors trigger intracellular biochemical signals by binding to cell membrane

receptors. In general, these membrane-bound receptors are protein kinases that

convert an extracellular signal into an intracellular signal. Many of the proteins that

participate in the intracellular signal transduction system are encoded by protooncogenes that are divided into subgroups based on their cellular location or

enzymatic function.

4 Oncogenes comprise a family of genes that result from gain of function mutations of

their normal counterparts, proto-oncogenes. The normal function of proto-oncogenes

is to stimulate proliferation in a controlled context. Activation of oncogenes can lead

to stimulation of cell proliferation and development of a malignant phenotype.

5 Tumor suppressor genes are involved in the development of most cancers and are

232usually inactivated in a two-step process in which both copies of the tumor

suppressor gene are mutated or inactivated by epigenetic mechanisms like

methylation. The most commonly mutated tumor suppressor gene in human cancers

is p53.

2 T lymphocytes have a central role in the generation of immune responses by acting as

helper cells in both humoral and cellular immune responses and by acting as effector

cells in cellular responses. T cells can be distinguished from other types of

lymphocytes by their cell surface phenotype, based on the pattern of expression of

various molecules, and by differences in their biologic functions.

2 There are two major subsets of mature T cells that are phenotypically and

functionally distinct: T-helper/inducer cells, which express the CD4 cell surface

marker, and the T-suppressor/cytotoxic cells, which express the CD8 marker. TH1

and TH2 are two helper T-cell subpopulations that control the nature of an immune

response by secreting a characteristic and mutually antagonistic set of cytokines:

Clones of TH1 produce interleukin-2 (IL-2) and interferon-γ (IFN-γ), whereas TH2

clones produce IL-4, IL-5, IL-6, and IL-10.

Advances in molecular biology and genetics have improved our understanding of

basic biologic concepts and disease development. The knowledge acquired with

the completion of the human genome project, the data available through The

Cancer Genome Atlas (TCGA), the development of novel diagnostic modalities,

such as the microarray technology for the analysis of DNA and proteins, and the

emergence of treatment strategies that target specific disease mechanisms all have

an increasing impact on the specialty of obstetrics and gynecology.

Normal cells are characterized by discrete metabolic, biochemical, and

physiologic mechanisms. Specific cell types differ with respect to their mainly

genetically determined responses to external influences (Fig. 6-1). An external

stimulus is converted to an intracellular signal, for example, via a cell membrane

receptor. The intracellular signal is transferred to the nucleus and generates

certain genetic responses that lead to changes in cellular function, differentiation,

and proliferation. Although specific cell types and tissues exhibit unique

functions and responses, many basic aspects of cell biology and genetics are

common to all eukaryotic cells.

CELL CYCLE

Normal Cell Cycle

Adult eukaryotic cells possess a well-balanced system of continuous

production of DNA (transcription) and proteins (translation). Proteins are

constantly degraded and replaced depending on the specific cellular

233requirements. Cells proceed through a sequence of phases called the cell

cycle, during which the DNA is distributed to two daughter cells (mitosis)

and subsequently duplicated (synthesis phase). This process is controlled at key

checkpoints that monitor the status of cells, for example, the amount of DNA

present in each cell. The cell cycle is regulated by a small number of

heterodimeric protein kinases that consist of a regulatory subunit (cyclin) and a

catalytic subunit (cyclin-dependent kinase). Association of a cyclin with a cyclindependent kinase (CdkC) determines which proteins will be phosphorylated at a

specific point during the cell cycle.

The cell cycle is divided into four major phases: M phase (mitosis), G1

phase (period between mitosis and initiation of DNA replication), S phase

(DNA synthesis), and G2 phase (period between completion of DNA synthesis

and mitosis) (Fig. 6-2). Post-mitotic cells can “exit” the cell cycle into the socalled G0 phase and remain for days, weeks, or even a lifetime without

further proliferation. The duration of the cell cycle may be highly variable,

although most human cells complete the cell cycle within approximately 24

hours. During a typical cell cycle, mitosis lasts about 30 to 60 minutes, the G1

phase 7 to 10 hours, S phase 10 hours, and G2 phase 5 hours. With respect to the

cell cycle, there are three subpopulations of cells:

FIGURE 6-1 External stimuli affect the cell, which has a specific coordinated response.

234FIGURE 6-2 The cell cycle.

1. Terminally differentiated cells cannot reenter the cell cycle.

2. Quiescent (G0) cells can enter the cell cycle if appropriately stimulated.

3. Dividing cells are currently in the cell cycle.

Red blood cells, striated muscle cells, uterine smooth muscle cells, and nerve

cells are terminally differentiated. Other cells, such as fibroblasts, exit from the

G1 phase into the G0 phase and are considered to be out of the cell cycle. These

235cells enter the cell cycle following exposure to specific stimuli, such as growth

factors and steroid hormones. Dividing cells are found in the gastrointestinal tract,

the skin, and the cervix.

G1 Phase

In response to specific external stimuli, cells enter the cell cycle by moving

from the G0 phase into the G1 phase. The processes during G1 phase lead to the

synthesis of enzymes and regulatory proteins necessary for DNA synthesis during

S phase and are mainly regulated by G1 cyclin-dependent kinase–cyclin

complexes (G1CdkC). Complexes of G1CdkC induce degradation of the S phase

inhibitors in late G1. Release of the S phase CdkC complex subsequently

stimulates entry into the S phase. Variations in the duration of the G1 phase of

the cell cycle, ranging from less than 8 hours to longer than 100 hours,

account for the different generation times exhibited by different types of

cells.

S Phase

The nuclear DNA content of the cell is duplicated during the S phase of the

cell cycle. The S-phase CdkC complex activates proteins of the DNA

prereplication complexes that assemble on DNA replication origins during G1.

The prereplication complex activates initiation of DNA replication and inhibits

the assembly of new prereplication complexes. This inhibition ascertains that each

chromosome is replicated only once during the S phase.

G2 Phase

RNA and protein synthesis occurs during the G2 phase of the cell cycle. The

burst of biosynthetic activity provides the substrates and enzymes to meet the

metabolic requirements of the two daughter cells. Another important event that

occurs during the G2 phase of the cell cycle is the repair of errors of DNA

replication that may have occurred during the S phase. Failure to detect and

correct these genetic errors can result in a broad spectrum of adverse

consequences for the organism and the individual cell (1). Defects in the DNA

repair mechanism are associated with an increased incidence of cancer. Mitotic

CdkC complexes are synthesized during the S and G2 phases, but are inactive

until DNA synthesis is completed.

M Phase

Nuclear–chromosomal division occurs during the mitosis or M phase. During

236this phase, the cellular DNA is equally distributed to each of the daughter cells.

Mitosis provides a diploid (2n) DNA complement to each somatic daughter cell.

Following mitosis, eukaryotic mammalian cells contain diploid DNA, reflecting a

karyotype that includes 44 somatic chromosomes and an XX or XY sex

chromosome complement. Exceptions to the diploid cellular content include

hepatocytes (4n) and the functional syncytium of the placenta.

Mitosis is divided into prophase, metaphase, anaphase, and telophase.

Mitotic CdkC complexes induce chromosome condensation during the prophase,

assembly of the mitotic spindle apparatus, and alignment of the chromosomes

during the metaphase. Activation of the anaphase promoting complex (APC)

leads to inactivation of the protein complexes that connect sister chromatids

during metaphase, permitting the onset of anaphase. During anaphase, sister

chromatids segregate to opposite spindle poles. The nuclear envelope breaks

down into multiple small vesicles early in mitosis and reforms around the

segregated chromosomes as they decondense during telophase. Cytokinesis is the

process of division of the cytoplasm that segregates the endoplasmic reticulum

and the Golgi apparatus during mitosis. After completion of mitosis, cells enter

the G1 phase and either reenter the cell cycle or remain in G0.

Ploidy

After meiosis, germ cells contain a haploid (1n) genetic complement. After

fertilization, a 46,XX or 46,XY diploid DNA complement is restored.

Restoration of the normal cellular DNA content is crucial to normal function.

Abnormalities of cellular DNA content cause distinct phenotypic

abnormalities, as exemplified by a hydatidiform molar pregnancy (see

Chapter 39). In a complete hydatidiform mole, an oocyte without any

nuclear genetic material (e.g., an empty ovum) is fertilized by one sperm. The

haploid genetic content of the fertilized ovum is duplicated, and the diploid

cellular DNA content is restored, resulting in a homozygous 46,XX gamete.

Less often, a complete hydatidiform mole results from the fertilization of an

empty ovum by two sperm, resulting in a heterozygous 46,XX or 46,XY gamete.

In complete molar pregnancies, the nuclear DNA is usually paternally derived,

embryonic structures do not develop, and trophoblast hyperplasia occurs. Rarely,

complete moles are biparental. This karyotype seems to be found in patients with

recurrent hydatidiform moles and is associated with a higher risk of persistent

trophoblastic disease.

A partial hydatidiform mole follows the fertilization of a haploid ovum by

two sperm, resulting in a 69 XXX, 69 XXY, or 69 XYY karyotype. A partial

mole contains paternal and maternal DNA, and both embryonic and placental

237development occur. Both the 69 YYY karyotype and the 46 YY karyotype are

incompatible with embryonic and placental development. These observations

demonstrate the importance of maternal genetic material, in particular the X

chromosome, in normal embryonic and placental development.

In addition to total cellular DNA content, the chromosome number is an

important determinant of cellular function. Abnormalities of chromosome

number, which often are caused by nondisjunction during meiosis, result in wellcharacterized clinical syndromes such as trisomy 21 (Down syndrome), trisomy

18, and trisomy 13.

Genetic Control of the Cell Cycle

Cellular proliferation must occur to balance normal cell loss and maintain

tissue and organ integrity. This process requires the coordinated expression of

many genes at discrete times during the cell cycle. In the absence of growth

factors, cultured mammalian cells are arrested in the G0 phase. With the addition

of growth factors, these quiescent cells pass through the so-called restriction point

14 to 16 hours later and enter the S phase 6 to 8 hours thereafter. The restriction

point or the G1/S boundary marks the point at which a cell commits to

proliferation. A second checkpoint is the G2/M boundary, which marks the

point at which repair of any DNA damage must be completed (2). To

successfully complete the cell cycle, a number of cell division cycle (cdc) genes

are activated.

Cell Division Cycle Genes

Among the factors that regulate the cell cycle checkpoints, proteins encoded

by the cdc2 family of genes and the cyclin proteins appear to play

particularly important roles (3). Growth factor–stimulated mammalian cells

express early- or delayed-response genes, depending on the chronologic sequence

of the appearance of specific RNAs. The early- and delayed-response genes act as

nuclear transcription factors and stimulate the expression of a cascade of other

genes. Early-response genes such as c-Jun and c-Fos enhance the transcription of

delayed-response genes such as E2Fs. E2F transcription factors are required for

the expression of various cell cycle genes and are functionally regulated by the

retinoblastoma (Rb) protein. Binding of Rb to E2F converts E2F from a

transcriptional activator to a repressor of transcription. Phosphorylation of Rb

inhibits its repressing function and permits E2F-mediated activation of genes

required for entry into the S phase. Cdk4-cyclin D, Cdk6-cyclin D, and Cdk2-

cyclin E complexes cause phosphorylation of Rb, which remains phosphorylated

throughout the S, G2, and M phases of the cell cycle. After completion of mitosis,

238a decline of the level of Cdk-cyclins leads to dephosphorylation of Rb by

phosphatases and, consequently, an inhibition of E2F in the early G1 phase.

Cdks are under evaluation as targets for cancer treatments because they are

frequently overactive in cancer disease and Cdk-inhibiting proteins are

dysfunctional. The Cdk4 inhibitor P1446A-05, for example, specifically inhibits

Cdk4-mediated G1-S phase transition, arresting cell cycling and inhibiting cancer

cell growth (4). SNS-032 selectively binds to Cdk2, -7, and -9, preventing their

phosphorylation and activation and subsequently preventing cell proliferation.

As cells approach the G1-S phase transition, synthesis of cyclin A is initiated.

The Cdk2-cyclin A complex can trigger initiation of DNA synthesis by

supporting the prereplication complex. The cyclin-dependent kinase 1 (Cdk1)

protein, formerly called p34 cdc2, and specific cyclins (namely cyclin B1) form a

complex heterodimer referred to as mitosis-promoting factor (5), which catalyzes

protein phosphorylation and drives the cell into mitosis. Cdk1 assembles with

cyclin A and cyclin B in the G2 phase and promotes the activity of the mitosis

promoting factor (MPF). Mitosis is initiated by activation of the cdc gene at the

G2-M checkpoint (6). After the G2-M checkpoint is passed, the cell undergoes

mitosis. In the presence of abnormally replicated chromosomes, progression past

the G2-M checkpoint does not occur.

The p53 tumor suppressor gene participates in cell cycle control. Cells

exposed to radiation therapy exhibit an S-phase arrest that is accompanied by

increased expression of p53. This delay permits the repair of radiation-induced

DNA damage. In the presence of p53 mutations, the S-phase arrest that normally

follows radiation therapy does not occur (7). The wild type p53 gene can be

inactivated by the human papillomavirus (HPV) E6 protein, preventing S-phase

arrest in response to DNA damage.

Apoptosis

[1] The regulation and maintenance of normal tissue requires a balance

between cell proliferation and programmed cell death, or apoptosis. When

proliferation exceeds programmed cell death, the result is hyperplasia. When

programmed cell death exceeds proliferation, the result is atrophy. Programmed

cell death is a crucial concomitant of normal embryologic development. This

mechanism accounts for deletion of the interdigital webs, palatal fusion, and

development of the intestinal mucosa (8). Programmed cell death is an important

phenomenon in normal physiology. The reduction in the number of endometrial

cells following alterations in steroid hormone levels during the menstrual cycle is,

in part, a consequence of programmed cell death (9). In response to androgens,

granulosa cells undergo programmed cell death (e.g., follicular atresia) (10).

239Programmed cell death, or apoptosis, is an energy-dependent, active

process that is initiated by the expression of specific genes. This process is

distinct from cell necrosis, although both mechanisms result in a reduction in the

total cell number. In programmed cell death, cells shrink and undergo

phagocytosis. Conversely, groups of cells expand and lyse when undergoing cell

necrosis. This process is energy independent and results from noxious stimuli.

Programmed cell death is triggered by a variety of factors, including intracellular

signals and exogenous stimuli such as radiation exposure, chemotherapy, and

hormones. Cells undergoing programmed cell death may be identified on the

basis of histologic, biochemical, and molecular biologic changes. Histologically,

apoptotic cells exhibit cellular condensation and fragmentation of the nucleus.

Biochemical correlates of impending programmed cell death include an increase

in transglutaminase expression and fluxes in intracellular calcium concentration

(11).

Programmed cell death emerged as an important factor in the growth of

neoplasms. Historically, neoplastic growth was characterized by uncontrolled

cellular proliferation that resulted in a progressive increase in tumor burden. It is

recognized that the increase in tumor burden associated with progressive

disease reflects an imbalance between cell proliferation and cell death. Cancer

cells fail to respond to the normal signals to stop proliferating, and they may fail

to recognize the physiologic signals that trigger programmed cell death.

MODULATION OF CELL GROWTH AND FUNCTION

The normal cell exhibits an orchestrated response to the changing extracellular

environment. The three groups of substances that signal these extracellular

changes are steroid hormones, growth factors, and cytokines. The capability to

respond to these stimuli requires a cell surface recognition system, intracellular

signal transduction, and nuclear responses for the expression of specific genes in

a coordinated fashion. [2] Among the genes that participate in the control of

cell growth and function, proto-oncogenes and tumor suppressor genes are

particularly important. More than 100 proto-oncogene products that contribute

to growth regulation have been identified (Table 6-1) (12). As a group, protooncogenes exert positive effects upon cellular proliferation. In contrast, tumor

suppressor genes exert inhibitory regulatory effects on cellular proliferation

(Table 6-2).

Steroid Hormones

Steroid hormones play a crucial role in reproductive biology and in general

physiology. Among the various functions, steroid hormones influence pregnancy,

240cardiovascular function, bone metabolism, and an individual’s sense of wellbeing. The action of steroid hormones is mediated via extracellular signals to the

nucleus to affect a physiologic response.

Estrogens exert a variety of effects on growth and development of different

tissues. The effects of estrogens are mediated via estrogen receptors (ER),

intracellular proteins that function as ligand-activated transcription factors

and belong to the nuclear receptor superfamily (13). Two mammalian ERs

have been identified: ERα and ERβ. The structure of both receptors is similar

and consists of 6 domains named A through F from the N- to C-terminus,

encoded by 8 to 9 exons (14). Domains A and B are located at the N-terminus and

contain an agonist-independent transcriptional activation domain (activation

function 1 [AF-1]). The C domain is a highly conserved central DNA-binding

domain composed of two zinc fingers through which ER interacts with the major

groove and the phosphate backbone of the DNA helix. The C-terminus of the

protein contains domains E and F and functions as ligand-binding domain (LBD–

domain E) and AF-2–domain F (Fig. 6-3).

Table 6-1 Proto-Oncogenes

Proto-Oncogenes Gene Product/Function

Growth Factors

Fibroblast growth factor

fgf-5

Sis Platelet-derived growth factor β

hst, int-2

Transmembrane Receptors

erbB Epidermal growth factor (EGF)

receptor

HER2/neu EGF-related receptor

Fms Colony-stimulating factor (CSF)

receptor

Kit Stem cell receptor

Trk Nerve growth factor receptor

241Inner-Membrane Receptor

bcl-2

H-ras, N-ras, K-ras

fgr, lck, src, yes

Cytoplasmic Messengers

Crk

cot, plm-1, mos, raf/mil

Nuclear DNA-Binding Proteins

erb-B1

jun, ets-1, ets-2, fos, gil-1, rel, ski, vav

lyl-1, maf, myb, myc, L-myc, N-myc,

evi-1

Table 6-2 Tumor Suppressor Genes

p53 Mutated in as many as 50% of solid tumors

Rb Deletions and mutations predispose to retinoblastoma

PTEN Dual specificity phosphatase that represses PI3-kinase/Akt pathway

activation with negative effect on cell growth

P16INK4a Binds to cyclin-CDK4 complex inhibiting cell cycle progression

FHIT Fragile histidine triad gene with tumor suppressor function via

unknown mechanisms

WT1 Mutations are correlated with the Wilms tumor

NF1 Neurofibromatosis gene

APC Associated with colon cancer development in patients with familial

adenomatous

Activation of transcription via the ER is a multistep process. The initial

step requires activation of the ER via various mechanisms (Fig. 6-4). For

242example, estrogens such as 17β-estradiol can diffuse into the cell and bind to the

LBD of the ER. Upon ligand binding, the ER undergoes conformational changes

followed by a dissociation of various bound proteins, mainly heat shock proteins

90 and 70 (Hsp90 and Hsp70). Activation of the ER also requires phosphorylation

by several protein kinases, including casein kinase II, PKA, and components of

the Ras/MAPK (mitogen-activated protein kinase) pathway (14). Four

phosphorylation sites of the ER are clustered in the NH2 terminus with the AF-1

region.

FIGURE 6-3 Structure of the two mammalian estrogen receptors. ERα (595 amino acids)

and ERβ (530 amino acids) consist of six domains (A to F from the N- to C-terminus).

Domains A and B at the N-terminus contain an agonist-independent transcriptional

activation domain (activation function 1, or AF-1). The C domain is the central DNAbinding sequence (DBD). Domains E and F function as the ligand-binding domain (LBD)

and activation function 2 (AF-2). Also shown is the structure of the ER ligand 17β-

estradiol.

243FIGURE 6-4 Activation of estrogen receptor–mediated transcription. Intracellular

estrogen receptor signaling is mediated via different pathways. A: 17β-Estradiol diffuses

through the cell membrane and binds to cytoplasmic ER. The ER is subsequently

phosphorylated, undergoes dimerization, and binds to the estrogen response element (ERE)

on the promoter of an estrogen responsive gene. B: Estrogen ligand binds to membranebound ER and activates the mitogen-activated protein kinase (MAPK) pathways that

support ER-mediated transcription. C: Binding of cytokines such as insulinlike growth

factor (IGF) or epidermal growth factor to their membrane receptor can cause activation of

protein kinases like PKA, which subsequently activates ER by phosphorylation.

The activated ER elicits a number of different genomic as well as

244nongenomic effects on intracellular signaling pathways. The classical steroid

signaling pathway involves binding of the activated ER to an estrogen responsive

element (ERE) on the genome as homodimers and subsequent stimulation of

transcription (15). The minimal consensus sequence for the ERE is a 13 bp

palindromic inverted repeat (IR) and is defined as 5′-GGTCAnnnTGACC-3′.

Genes that are regulated by activated ERs include early gene responses such as cmyc, c-fos, and d-jun, and genes encoding for growth factors such as insulin

growth factor (IGF-1 and IGF-2), epidermal growth factor (EGF), transforming

growth factor α (TGF-α), and colony-stimulating factor (CSF-1).

Various ligands with different affinities to the ER were developed and are

called selective estrogen receptor modulators (SERMs). Tamoxifen, for

example, is a mixed agonist/antagonist for ERα, but it is a pure antagonist

for ERβ. The ERβ receptor is ubiquitously expressed in hormone-responsive

tissues, whereas the expression of ERα fluctuates in response to the hormonal

milieu. The cellular and tissue effects of an estrogenic compound appear to reflect

a dynamic interplay between the actions of these ER isoforms. These observations

underscore the complexity of estrogen interactions with both normal and

neoplastic tissues. Mutations of hormone receptors and their functional

consequences illustrate their important contributions to normal physiology.

Growth Factors

Growth factors are polypeptides that are produced by a variety of cell types

and exhibit a wide range of overlapping biochemical actions. Growth factors

bind to high-affinity cell membrane receptors and trigger complex positive and

negative signaling pathways that regulate cell proliferation and differentiation

(16). In general, growth factors exert positive or negative effects upon the cell

cycle by influencing gene expression related to events that occur at the G1-S

cell cycle boundary.

Because of their short half-life in the extracellular space, growth factors

act over limited distances through autocrine or paracrine mechanisms. In the

autocrine loop, the growth factor acts on the cell that produced it. The paracrine

mechanism of growth control involves the effect of growth factors on another cell

in proximity. Growth factors that play an important role in female reproductive

physiology are listed in Table 6-3. The biologic response of a cell to a specific

growth factor depends on a variety of factors, including the cell type, the cellular

microenvironment, and the cell cycle status.

The regulation of ovarian function occurs through autocrine, paracrine,

and endocrine mechanisms. The growth and differentiation of ovarian cells are

particularly influenced by the insulinlike growth factors (IGF) (Fig. 6-5). IGFs

245amplify the actions of gonadotropin hormones on autocrine and paracrine growth

factors found in the ovary. IGF-1 acts on granulosa cells to cause an increase in

cAMP, progesterone, oxytocin, proteoglycans, and inhibin. On theca cells, IGF-1

causes an increase in androgen production. Theca cells produce tumor necrosis

factor α (TNF-α) and EGF, which are regulated by follicle-stimulating hormone

(FSH). EGF acts on granulosa cells to stimulate mitogenesis. Follicular fluid

contains IGF-1, IGF-2, TNF-α, TNF-β, and EGF. Disruption of these autocrine

and paracrine intraovarian pathways may be the basis of polycystic ovarian

disease, disorders of ovulation, and ovarian neoplastic disease.

TGF-β activates intracytoplasmic serine threonine kinases and inhibits cells in

the late G1 phase of the cell cycle (17). It appears to play an important role in

embryonic remodeling. Mullerian-inhibiting substance (MIS), which is

responsible for regression of the mullerian duct, is structurally and functionally

related to TGF-β (18). TGF-α is an EGF homologue that binds to the EGF

receptor and acts as an autocrine factor in normal cells. As with EGF, TGF-α

promotes the entry of G0 cells into the G1 phase of the cell cycle. The role of

growth factors in endometrial growth and function was the subject of several

reviews (17,19). Similar to the ovary, autocrine, paracrine, and endocrine

mechanisms of control also occur in the endometrial tissue.

Table 6-3 Growth Factors That Play Important Roles in Female Reproductive

Physiology

246Intracellular Signal Transduction

[3] Growth factors trigger intracellular biochemical signals by binding to cell

membrane receptors. In general, these membrane-bound receptors are

protein kinases that convert an extracellular signal into an intracellular

signal. The interaction between growth factor ligand and its receptor results in

receptor dimerization, autophosphorylation, and tyrosine kinase activation.

Activated receptors in turn phosphorylate substrates in the cytoplasm and trigger

the intracellular signal transduction system (Fig. 6-6). The intracellular signal

transduction system relies on serine threonine kinases, src-related kinases, and G

proteins. Intracellular signals activate nuclear factors that regulate gene

expression. Many of the proteins that participate in the intracellular signal

transduction system are encoded by proto-oncogenes that are divided into

subgroups based on their cellular location or enzymatic function (Fig. 6-7)

(20).

247FIGURE 6-5 The regulation of ovarian function occurs through autocrine, paracrine, and

endocrine mechanisms.

248249FIGURE 6-6 Pathways of intracellular signal transduction.

The raf and mos proto-oncogenes encode proteins with serine threonine kinase

activity. These kinases integrate signals originating at the cell membrane with

those that are forwarded to the nucleus (21). Protein kinase C (PKC) is an

important component of a second messenger system that exhibits serine threonine

kinase activity. This enzyme plays a central role in phosphorylation, which is a

general mechanism for activating and deactivating proteins. It also plays an

important role in cell metabolism and division (22).

The Src family of tyrosine kinases is related to PKC and includes protein

products encoded by the src, yes, fgr, hck, lyn, fyn, lck, alt, and fps/fes protooncogenes. These proteins bind to the inner cell membrane surface.

The G proteins are guanyl nucleotide-binding proteins. The heterotrimeric, or

large G proteins, link receptor activation with effector proteins such as adenyl

cyclase, which activates the cAMP-dependent, kinase-signaling cascade (23). The

monomeric or small G proteins, encoded by the Ras proto-oncogene family, are

designated p21 and are particularly important regulators of mitogenic signals. The

p21 Ras protein exhibits guanyl triphosphate (GTP) binding and GTPase activity.

Hydrolysis of GTP to guanyl diphosphate (GDP) terminates p21 Ras activity. The

p21 Ras protein influences the production of deoxyguanosine and inositol

phosphate (IP) 3, arachidonic acid production, and IP turnover.

The phosphoinositide 3 (PI3) kinase can be activated by various growth factors

like platelet-derived growth factor (PDGF) or IGF results. Activation of PI3

kinase results in an increase of intracellular, membrane-bound lipids,

phosphatidylinositol-(3),(4)-diphosphate (PIP2), and phosphatidylinositol-(3),(4),

(5)-triphosphate (PIP3). The Akt protein is subsequently phosphorylated by PIP3-

dependent kinases (PDK) for full activation. Activated Akt is released from the

membrane and elicits downstream effects that lead to an increase in cell

proliferation, prevention of apoptosis, invasiveness, drug resistance, and

neoangiogenesis (24). The PTEN (phosphatase and chicken tensin homologue

deleted on chromosome 10) protein is an important factor in the PI3 kinase

pathway, because it counteracts the activation of Akt by dephosphorylating PIP3.

Cells with mutated tumor suppressor gene PTEN and lack of functional PTEN

expression display an increased proliferation rate and decreased apoptosis,

possibly supporting the development of a malignant phenotype. PTEN frequently

is mutated in endometrioid adenocarcinoma. Furthermore, lack of functional

PTEN expression was described in endometriosis.

The mammalian target of rapamycin (mTOR) is regulated by the PI3 kinase

pathway. mTOR is a serine/threonine protein kinase that regulates a variety of

250cellular processes, including proliferation, motility, and translation (25). mTOR

integrates the input from various upstream pathways, including insulin and

growth factors like IGF proteins. The mTOR pathway provides important survival

signals for cancer cells and therefore was one focus of targeted drug development

(26). For example, rapamycin inhibits mTOR by associating with its intracellular

receptor FKBP12. Derivatives of rapamycin like everolimus (RAD001) and

temsirolimus (CCI779) showed promising results in clinical trials (27).

FIGURE 6-7 Proto-oncogenes are divided into subgroups based on their cellular location

or enzymatic function.

mTOR functions as the catalytic subunit of two different protein complexes.

Among the proteins associated with mTOR complex 1 (mTORC1) is mTOR, the

regulatory associated protein of mTOR (Raptor), and PRAS40. This complex

functions as a nutrient and energy sensor and controls protein synthesis (28).

mTORC1 is activated by insulin, growth factors, amino acids and oxidative stress,

low nutrient levels, reductive stress, and growth factor deprivation inhibit its

activity. In contrast, the mTOR complex 2 (mTORC2) contains, among others,

251mTOR, the rapamycin-insensitive protein Rictor, and mammalian stress-activated

protein kinase interacting protein 1 (mSIN1). mTORC2 regulates the cytoskeleton

and phosphorylates Akt (29). Its regulation is complex, but involves insulin,

growth factors, serum, and nutrient levels.

Expression of Genes and Proteins

Ovarian, endometrial, and cervical cancers have distinct molecular and genomic

profiles. Ovarian carcinomas, in particular the high-grade serous histologic

subtypes, are characterized by genomic instability with low frequencies of

conserved mutations (30). Despite substantial differences between the

gynecologic cancers, activation of the PI3K pathway is common.

Endometrial cancers demonstrate the highest frequency of PI3K pathway

alterations (31). The rate of alterations in PIK3CA range from 25% to 40% and in

the PIK3R1 from 15% to 25% (32). In addition to PI3K alterations, 30% to 90%

of endometrial cancers have impaired PTEN function, most often identified in

endometrioid histologies (33). PI3K inhibition may impair proliferation of

endometrial cancer cells. In addition, loss of PTEN or PIK3CA mutation may

serve as biomarkers associated with response to novel targeted agents that

antagonize PI3K (34). Studies utilizing patient-derived xenograft (PDX) models

of endometrial carcinomas, with and without alterations in PIK3CA, have

suggested that pan-PI3K inhibition may produce significant anti-tumor responses.

Most of the published data regarding PI3K pathway inhibitors in endometrial

cancer have been based on mTOR inhibition. Rapalog inhibitors (everolimus,

ridaforolimus, and temsirolimus) have been demonstrated to inhibit mTORC1

complex kinase activity. However, in clinical application, the response rates to

monotherapy have been low, ranging from 4% to 25%; the higher response rates

correlating with patients extensively pretreated with cytotoxic chemotherapy (35).

Combination treatment of everolimus and letrozole showed a 32% response rate

(11 of 35 patients) (36). These response rates and duration are similar to treatment

with alternating courses of megestrol acetate and tamoxifen, with an overall RR

of 27%. Based on these comparable response findings, a randomized study is

underway comparing these two treatment strategies (GOG-3007) (36).

Genetic alterations of the PI3K pathway have been identified in up to 70% of

ovarian cancers (37). Gain of function mutations in PIK3CA have been observed

in 4% to 12% of ovarian cancers (38). Ovarian clear cell and endometrioid

adenocarcinomas demonstrate a higher frequency of mutations with 33% to 40%

activating mutations in PIK3CA in clear cell carcinomas, and 12% to 20% of

endometrioid carcinomas (39).

In cervical cancer, amplifications and activating mutations of the PIK3CA gene

have been demonstrated in 23% to 36% of cases, with the highest mutation rate

252found in PIK3CA gene (40). The HPV oncogenic proteins, namely E6, and E7

enhance PI3K pathway signaling via direct activation of PI3K, as well as

downstream AKT and rpS6K. These proteins induce ligand-dependent signaling

indirectly through epidermal growth factor receptor (EGFR). Similar to ovarian

cancer, data pertaining to the use of targeted PI3K pathway therapy in cervical

carcinoma is limited, with studies underway.

Regulation of genetic transcription and replication is crucial to the normal

function of the daughter cells, the tissues, and ultimately the organism.

Transmission of external signals to the nucleus by way of the intracellular

signal transduction cascade culminates in the transcription of specific genes

and translation of the mRNA into proteins that ultimately affect the

structure, function, and proliferation of the cell.

The human genome project resulted in the determination of the sequence

of DNA of the entire human genome (41). With the completion of this

project, it appears that the human haploid genome contains 23,000 protein

coding genes. Sequencing the human genome is a major scientific achievement

that has opened the door for more detailed studies of structural and functional

genomics. Structural genomics involves the study of three-dimensional

structures of proteins based on their amino acid sequences. Functional

genomics provides a way to correlate the structure and function. Proteomics

involves the identification and cataloging of all proteins used by a cell, and

cytomics involves the study of cellular dynamics, including intracellular

system regulation and response to external stimuli. The transcriptome is the

set of all RNA molecules, including mRNA, rRNA, tRNA, and other noncoding

RNA produced in one or a population of cells. The transcriptome varies with

external environmental conditions and reflects the actively expressed genes. The

metabolome describes a set of small-molecule metabolites, including

hormones and signaling molecules, that are found in a single organism.

Similar to the transcriptome and proteome, the metabolome is subject to

rapid changes (42). The kinome of an organism describes a set of protein

kinases, enzymes that are crucial for phosphorylation reactions.

Cancer Genetics

Cancer is a genetic disease that results from a series of mutations in various

cancer genes. Uncontrolled cell growth occurs because of accumulation of

somatic mutations or the inheritance of one or more mutations through the

germline, followed by additional somatic mutations. The mutation in genes

that are directly involved in normal cellular growth and proliferation can

lead to the development of uncontrolled growth, invasion, and metastasis.

According to the Knudson hypothesis, which was first described in

253children with hereditary retinoblastoma, two hits or mutations within the

genome of a cell are required for a malignant phenotype to develop (43). In

hereditary cancers, the first hit is present in the genome of every cell. Only

one additional hit is necessary, therefore, to disrupt the correct function of

the second cancer gene allele. In contrast, sporadic cancers develop in cells

without hereditary mutations in the cancer predisposing alleles. In this case,

both hits must occur in a single somatic cell to disrupt both cancer gene

alleles (Fig. 6-8).

Most adult solid tumors require 5 to 10 rate-limiting mutations to acquire

the malignant phenotype. Among these mutations, some are responsible for

causing the cancer phenotype, whereas others might be considered bystander

mutations, as with, for example, the amplification of genes that are adjacent to an

oncogene. The most compelling evidence for the mutagenic tumor development

process is that the age-specific incidence rates for most human epithelial tumors

increase at roughly the fourth to eighth power of elapsed time.

Gatekeepers and Caretakers

[4] Cancer susceptibility genes are divided into “gatekeepers” and

“caretakers” (44). Gatekeeper genes control cellular proliferation and are

divided into oncogenes and tumor suppressor genes. In general, oncogenes

stimulate cell growth and proliferation, and tumor suppressor genes reduce

the rate of cell proliferation or induce apoptosis. Gatekeepers prevent the

development of tumors by inhibiting growth or promoting cell death.

Examples of gatekeeper genes include the tumor suppressor gene p53 and the

retinoblastoma gene.

Caretaker genes preserve the integrity of the genome and are involved in DNA

repair (stability genes). The inactivation of caretakers increases the likelihood of

persistent mutations in gatekeeper genes and other cancer-related genes. The

DNA mismatch repair (MMR) genes, MLH1, MSH2, and MSH6, are examples of

caretaker genes.

254FIGURE 6-8 Hereditary and sporadic cancer development based on the Knudson “twohit” genetic model. All cells harbor one mutant tumor suppressor gene allele in hereditary

cancer. The loss of the second allele results in the malignant phenotype. Sporadic cancers

develop in cells with normal genome, therefore requiring both alleles to be inactivated (two

hits).

Hereditary Cancer

Most cancers are caused by spontaneous somatic mutations. However, a

small percentage of cancers arise on a heritable genomic background. About

12% of all ovarian cancers and about 5% of endometrial cancers are

considered to be hereditary (45,46). Germline mutations require additional

mutations at one or more loci for tumorigenesis to occur. These mutations occur

via different mechanisms, for example, via environmental factors such as ionizing

radiation or mutations of stability genes. Characteristics of hereditary cancers

include diagnosis at a relatively early age and a family history of cancer,

usually of a specific cancer syndrome, in two or more relatives. Hereditary

cancer syndromes associated with gynecologic tumors are summarized in Table

6-4.

[4] Various cancer-causing genetic and epigenetic mechanisms are described.

On the genomic level, gain of function gene mutations can lead to a

conversion of proto-oncogenes into oncogenes, and loss of function gene

mutations can inactivate tumor suppressor genes. Epigenetic changes include

DNA methylation, which can cause inactivation of tumor suppressor gene

expression by preventing the correct function of the associated promoter

sequence. Collectively, these genetic and epigenetic changes are responsible for

the development of cancer characterized by the ability of cells to invade and

metastasize, grow independently of growth factor support, and escape from

255antitumor immune responses.

Oncogenes

Oncogenes comprise a family of genes that result from the gain of function

mutations of their normal counterparts, proto-oncogenes. The normal

function of proto-oncogenes is to stimulate proliferation in a controlled

context. Activation of oncogenes can lead to stimulation of cell proliferation

and development of a malignant phenotype. Oncogenes were initially

discovered through retroviral tumorigenesis. Viral infection of mammalian cells

can result in integration of the viral sequences into the proto-oncogene sequence

of the host cell. The integrated viral promoter activates transcription from the

surrounding DNA sequences, including the proto-oncogene. Enhanced

transcription of the proto-oncogene sequences results in the overexpression of

growth factors, growth factor receptors, and signal transduction proteins, which

result in stimulation of cell proliferation. One of the most important group of viral

oncogenes is the family of ras genes, which include c-H(Harvey)-ras, cK(Kirsten)-ras, and N(Neuroblastoma)-ras.

Tumor Suppressor Genes

[5] Tumor suppressor genes are involved in the development of most cancers

and are usually inactivated in a two-step process in which both copies of the

tumor suppressor gene are mutated or inactivated by epigenetic mechanisms

like methylation (47). The most commonly mutated tumor suppressor gene in

human cancers is p53 (48). The p53 protein regulates transcription of other

genes involved in cell cycle arrest such as p21. Upregulation of p53 expression is

induced by DNA damage and contributes to cell cycle arrest, allowing DNA

repair to occur. p53 also plays an important role in the initiation of apoptosis. The

most common mechanism of inactivation of p53 differs from the classic two-hit

model. In most cases, missense mutations that change a single amino acid in the

DNA-binding domain of p53 result in overexpression of nonfunctional p53

protein in the nucleus of the cell.

The identification of tumor suppressor genes was facilitated by positional

cloning strategies. The main approaches are cytogenetic studies to identify

chromosomal alterations in tumor specimens, DNA linkage techniques to localize

genes involved in inherited predisposition to cancer, and examination for loss of

heterozygosity (LOH) or allelic alterations among studies in sporadic tumors.

Comparative genomic hybridization (CGH) allows fluorescence identification of

chromosome gain and loss in human cancers within a similar experiment.

Table 6-4 Hereditary Cancer Syndromes Associated With Gynecologic Tumors

256Hereditary

Syndrome

Gene

Mutation

Tumor Phenotype

Li–Fraumeni

syndrome

TP53,

CHEK2

Breast cancer, soft tissue sarcoma, adrenal

cortical carcinoma, brain tumors

Cowden

syndrome,

Bannayan–

Zonana

syndrome

PTEN Breast cancer, hamartoma, glioma,

endometrial cancer

Hereditary

breast and

ovarian cancer

BRCA1,

BRCA2

Cancer of breast, ovary, fallopian tube

Hereditary

nonpolyposis

colorectal

cancer

(HNPCC)

MLH1,

MSH2,

MSH3,

MSH6,

PMS2

Cancer of colon, endometrium, ovary,

stomach, small bowel, urinary tract

Multiple

endocrine

neoplasia type

I

Menin Cancer of thyroid, pancreas, and pituitary,

ovarian carcinoid

Multiple

endocrine

neoplasia type

II

RET Cancer of thyroid and parathyroid,

pheochromocytoma, ovarian carcinoid

Peutz–Jeghers

syndrome

STK11 Gastrointestinal hamartomatous polyps,

tumors of the stomach, duodenum, colon,

ovarian sex cord tumor with annular tubules

(SCTAT)

Stability Genes

The third class of cancer genes is “stability genes,” which promotes

tumorigenesis in a different way from tumor suppressor genes or amplified

oncogenes. The main function of stability genes is the preservation of the

correct DNA sequence during DNA replication (caretaker function) (49).

Mistakes that are made during normal DNA replication or induced by exposure to

mutagens can be repaired by a variety of mechanisms that involve MMR genes,

257nuclear-type excision repair genes, and base excision repair genes. The

inactivation of stability genes potentially leads to a higher mutation rate in all

genes. However, only mutations in oncogenes and tumor suppressor genes

influence cell proliferation and confer a selective growth advantage to the mutant

cell. Similar to tumor suppressor genes, both alleles of stability genes must be

activated to cause loss of function.

Genetic Aberrations

Gene replication, transcription, and translation are imperfect processes, and

the fidelity is less than 100%. Genetic errors may result in abnormal structure

and function of genes and proteins. Genomic alterations such as gene

amplification, point mutations, and deletions or rearrangements were identified in

premalignant, malignant, and benign neoplasms of the female genital tract (Fig.

6-9).

258FIGURE 6-9 Genes can be amplified or undergo mutation, deletion, or rearrangement.

Amplification

Amplification refers to an increase in the copy number of a gene.

Amplification results in enhanced gene expression by increasing the amount of

template DNA that is available for transcription. Proto-oncogene amplification is

a relatively common event in malignancies of the female genital tract. The

HER2/neu proto-oncogene, also known as c-erbB-2 and HER2, encodes a 185

kDa transmembrane glycoprotein with intrinsic tyrosine kinase activity. It belongs

to a family of transmembrane receptor genes that includes the EGF receptors

erbB-1, erbB-3, and erbB-4. HER2/neu interacts with a variety of different

259cellular proteins that increase cell proliferation. Overexpression of HER2/neu was

demonstrated in about 30% of breast cancers, 20% of advanced ovarian cancers,

and as many as 50% of endometrial cancers (50). The GOG-177 study (a

randomized prospective phase III study including patients with measurable stages

III to IV, or recurrent endometrial carcinoma and randomly assigned treatment

with either doxorubicin and cisplatin or doxorubicin, cisplatin, and paclitaxel

with G-CSF) also included tumor collection within the study parameters. A

review of the tumor specimens showed a 44% rate of HER2 overexpression (2+

or 3+ by immunohistochemical [IHC] staining) and 12% HER2 amplification by

fluorescence in situ hybridization (FISH) (51). Morrison et al. investigated HER2

expression and amplification in women with endometrial cancer, including a wide

variety of histologies. The investigation showed a correlation between HER2

expression and amplification and tumors of higher grade/stage, lymph node

positivity, and survival outcomes. Overall survival was significantly shorter in

patients with HER2 expression (median 5.2 years) and/or showed amplification

(median 3.5 years) versus those that did not (median 13 years) (52). The clinical

significance of HER2 overexpression or gene amplification continues to be

debated.

Point Mutations

Point mutations of a gene may remain without any consequence for the

expression and function of the protein (gene polymorphism). However, point

mutations can alter a codon sequence and subsequently disrupt the normal

function of a gene product. The ras gene family is an example of oncogeneencoded proteins that disrupt the intracellular signal transduction system

following point mutations. Transforming Ras proteins contain point mutations in

critical codons (i.e., codons [11, 12], [59], [61]) with decrease of GTPase activity

and subsequent expression of constitutively active Ras. Point mutations of the p53

gene are the most common genetic mutations described in solid tumors. These

mutations occur at preferential “hot spots” that coincide with the most highly

conserved regions of the gene. The p53 tumor suppressor gene encodes for a

phosphoprotein that is detectable in the nucleus of normal cells. When DNA

damage occurs, p53 can arrest cell cycle progression to allow the DNA to be

repaired or undergo apoptosis. The lack of function of normal p53 within a cancer

cell results in a loss of control of cell proliferation with inefficient DNA repair

and genetic instability. Somatic mutations of the p53 gene occur in approximately

50% of advanced ovarian cancers and 30% to 40% of endometrial cancers

(predominantly serous histology) but are uncommon in cervical cancer (53,54).

BRCA

260Point mutations in the BRCA1 and BRCA2 genes can alter the activity of

these genes and predispose to the development of breast and ovarian

cancer(s) (55). The frequency of BRCA1 and BRCA2 mutations in the general

population in the United States is estimated at 1:250. Specific founder mutations

were reported for various ethnic groups. For example, two BRCA1 mutations

(185delAG and 5382insC) and one BRCA2 mutation (6174delT) are found in

2.5% of Ashkenazi Jews of Central and Eastern European descent. Additional

founder mutations were described in other ethnic groups, including from the

Netherlands (BRCA1, 2804delAA and several large deletion mutations), Iceland

(BRCA2, 995del5), and Sweden (BRCA1, 3171ins5).

The BRCA proteins are involved in DNA repair. If DNA is damaged, for

example, by ionizing radiation or chemotherapy, the BRCA2 protein binds to the

RAD51 protein, which is central for the repair of double-stranded breaks via

homologous recombination. BRCA2 regulates the availability and activity of

RAD51 in this key reaction. Phosphorylation of the BRCA2/RAD51 complex

allows RAD51 to bind to the site of DNA damage and, in conjunction with

several other proteins, mediates repair of DNA by homologous recombination.

BRCA1 functions within a complex network of protein–protein interactions,

mediating DNA repair by homologous recombination and regulating transcription

via the BRCA1-associated surveillance complex (BASC).

Deletions and Rearrangements

Individuals carrying a germline BRCA1 or BRCA2 pathogenic mutation have an

elevated lifetime risk of developing breast and ovarian cancer(s). Based on a

meta-analysis by Chen et al., the 70-year risk of developing ovarian cancer is

40% and 18% for BRCA1 and BRCA2, respectively, and breast cancer is 57%

and 49% for BRCA1 and BRCA2, respectively (56). These germline mutations

are inherited in an autosomal-dominant fashion. Approximately 5% of breast and

20% of ovarian cancers arise in women carrying heterozygous germline mutations

in BRCA1 or BRCA2 (57). BRCA1 and BRCA2 are tumor suppressor genes.

Loss of nonmutated (wild-type) allele at either BRCA1 or BRCA2 locus, termed

locus-specific LOH, can be observed in tumors. Cells with loss of BRCA function

and resultant homologous recombination (HR)–based DNA repair deficiency

have enhanced sensitivity to DNA-damaging agents, namely cytotoxic platinumbased chemotherapies and targeted therapies (specifically inhibition of poly

[ADP-ribose] polymerases [PARPs]) (58). Although much research has

demonstrated that germline pathogenic mutations in BRCA1 or BRCA2 in

ovarian cancer can downregulate HR, and thereby increase a patient’s sensitivity

and response to platinum-based chemotherapy and PARP inhibition, there is less

evidence to support the potential impact of somatic sequence variants of BRCA

261function. Published studies have demonstrated that germline and somatic

pathogenic mutations in HR genes (BRCA1, BRCA2, ATM, BARD1, BRIP1,

CHEK1, CHEK2, FAM175A, MRE11A, NBN, PALB2, RAD51C, and RAD51D)

occur in 31% of patients with serous or nonserous ovarian cancer (59). Of these,

75% of germline HR mutations and 71% of somatic HR mutations were in BRCA

(60). The role of somatic pathogenic BRCA mutations in platinum-based

chemotherapy and PARP inhibitor sensitivity indicates that the number of

individuals with ovarian cancer who may benefit from such treatments are greater

than predicted by the frequency of germline pathogenic mutations alone.

Poly (ADP-Ribose) Polymerases

PARPs are vital components of DNA damage and repair pathways. The concept

of synthetic lethality is based on the idea that simultaneous loss of function of two

or more gene products can cause cell death, even if a deficiency in one is not

lethal (61). For example, a tumor cell without a lethal inactivating mutation of

BRCA DNA repair genes, might still be destroyed if also exposed to effects of

PARP inhibition. Given that HGSCs are associated with germline BRCA1 or

BRCA2 mutations, they remain the most suitable candidates for PARP inhibition.

DNA repair function is inherently decreased in tumor cells, compared with

normal cells. This discrepancy contributes to the high selectivity of PARP

inhibitors (61).

Three different PARP inhibitors have been approved by the FDA for the

treatment of ovarian cancer patients, each with a different clinical indication.

Olaparib and niraparib are indicated in patients with platinum-sensitive ovarian

cancer as maintenance therapy following response to platinum-based

chemotherapy for recurrent disease. Neither drug requires the presence of

germline or somatic mutations in BRCA1 or BRCA2 genes for this indication.

For the treatment of recurrent ovarian cancer with olaparib, patients must have

had three or more lines of chemotherapy and have a germline mutation in BRCA1

or BRCA2. Response rates in this patient population is about 30% based on

clinical trial data (62). Rucaparib is likewise approved for maintenance therapy in

patients with recurrent, platinum-sensitive ovarian cancer but requires the

presence of a germline or somatic BRCA1 or BRCA2 mutation (63). The most

common side effects with PARP inhibitors include hematologic toxicities

including thrombocytopenia and anemia, gastrointestinal side effects, in particular

nausea and fatigue.

Lynch Syndrome (Hereditary Nonpolyposis Colorectal Cancer Syndrome, HNPCC)

Lynch syndrome (LS) is an autosomal-dominant inherited disorder caused by

germline mutations in DNA MMR genes. Mutations are characterized by

262predisposition for cancers arising from the colon, rectum, small bowel,

endometrium, ovary, stomach, pancreas, renal pelvis, ureter, and brain (64). This

syndrome was first identified in patients with familial predisposition to

gastrointestinal cancers. Endometrial cancer often precedes colorectal and other

LS-associated malignancies. Women with LS have a 15% to 70% lifetime risk of

endometrial cancer and a 6% to 8% lifetime risk of ovarian cancer, with this wide

range accounting for a variety of histopathologic subtypes (65,66). Such a high

incidence has led to increased screening of endometrial carcinoma specimens for

abnormalities in the DNA MMR pathway. The most common and convincing

method of germline mutation analysis is focused on the four primary MMR genes

associated in LS, including MLH1, MSH2, MSH6, and PMS1. Mutations in

MSH2 and/or MLH1 account for 90% of heterozygous mutations, while MSH6

mutations account for most of the remaining cases (67). A smaller subset of

patients may carry mutations in the EPCAM gene, which can lead to Lynch

phenotype by way of hypermethylation and inactivation of MSH2 promoter. IHC

staining for loss of expression serves as the standard screening approach.

Microsatellite instability (MSI) testing is available as a substitute or additional

modality for abnormalities of the MMR system; however, this testing has been

shown to be less sensitive than IHC, primarily because of a failure to detect

several MSH6 germline mutation carriers (64).

Deletions and rearrangements reflect gross changes in the DNA template

that may result in the synthesis of a markedly altered protein product.

Somatic mutations may involve chromosomal translocations that result in

chimeric transcripts with juxtaposition of one gene to the regulatory region of

another gene. This mutation type is most commonly reported in leukemias,

lymphomas, and mesenchymal tumors. The Philadelphia chromosome in chronic

myeloid leukemia (CML), for example, is the result of a reciprocal translocation

between one chromosome 9 and one chromosome 22. The DNA sequence

removed from chromosome 9 contains the proto-oncogene c-ABL and inserts into

the BCR gene sequence on chromosome 22 (Philadelphia chromosome). The

resulting chimeric BCR-ABL gene product functions as a constitutively active

tyrosine kinase and stimulates cellular proliferation by such mechanisms as an

increase of growth factors.

Single-nucleotide polymorphism (SNP) describes a variation in the DNA

sequence (68). Single nucleotides in the genome differ between paired

chromosomes in either one individual or between two individuals. For example,

the sequences TGACTA and TCACTA contain one single change in the second

nucleotide from guanine (G) to cytosine (C). This results in a G and C allele for

this particular gene sequence. SNPs can occur within coding or noncoding

sequences of genes or in the intergenic regions. SNPs might not change the amino

263acid sequence of the protein that is produced (synonymous SNP) or produce a

different peptide (nonsynonymous SNPs). If SNPs are located in noncoding

regions, various other processes like gene splicing or transcription factor binding

might be affected.

The frequency of SNPs in a given population is provided by the minor allele

frequency. This frequency differs between ethnic groups and geographic

locations. SNPs were associated with various human diseases including cancer.

They influence the effect of drug treatment and responses to pathogens and

chemicals (69). SNPs are important for the comparison between genomes of

different populations, for example, providing information about the susceptibility

of a certain population to develop specific cancers (70).

THE CANCER GENOME ATLAS PROJECT

In 2006, the National Cancer Institute and the National Human Genome Research

Institute initiated TCGA project. The goal of the project is to provide a

comprehensive genomic characterization and sequence analysis of cancer

diseases. The initial phase included glioblastoma multiforme, lung, and ovarian

cancers (71,72).

TCGA is taking advantage of high-throughput genome analysis techniques,

including gene expression profiling, SNP genotyping, copy number variation

profiling, genomewide methylation profiling, microRNA profiling, and exon

sequencing (73). These data are accessible for researchers via TCGA webpage

(https://gdc.cancer.gov). The findings as pertaining to gynecologic malignancies

are summarized below.

Endometrial Carcinoma

An integrated genomic, transcriptomic, and proteomic characterization of 373

endometrial carcinomas (tumor samples and corresponding germline DNA) using

array- and sequencing-based technologies was performed (74). Uterine serous

tumors and 25% of high-grade endometrioid tumors exhibited extensive copy

number alterations, few DNA methylation changes, low ER and progesterone

receptor levels, and frequent TP53 mutations. The majority of endometrioid

tumors were identified as having frequent mutations in PTEN, CTNNB1,

PIK3CA, ARID1A, and KRAS, novel mutations in SWI/SNF chromatin

remodeling complex gene ARID5B, and few copy number alterations or TP53

mutations. In addition, 10% of endometrioid tumors were identified to have

markedly increased transversion mutation frequency and a newly identified

mutation in POLE. POLE is a catalytic subunit of DNA polymerase epsilon

involved in nuclear DNA replication and repair (75). These new findings classify

264endometrial carcinomas into four distinct categories: POLE ultramutated, MSI

hypermutated, copy-number low, and copy-number high. Somatic copy number

alterations (SCNAs) showed that most endometrioid tumors have few SCNAs,

most serous and serous-like tumors exhibit extensive SCNAs, and the extent of

SCNA roughly correlates with progression-free survival (74).

According to TCGA data, endometrial cancer has higher-frequency mutations

in the PI(3)K/AKT pathway than any other tumor type that has been studied.

Endometrioid endometrial carcinomas demonstrate parallels with characteristics

of colorectal carcinoma, specifically high frequency of MSI (40% and 11%,

respectively), POLE mutations (7% and 3%, respectively) which lends to

ultrahigh mutation rates, and frequent activation of WNT/CTNNB1 signaling.

Yet, endometrial carcinomas are unlike those of colorectal carcinomas as they

have unique KRAS and CTNNB1 mutations and a distinct means by which

pathway activation is achieved. Data pertaining to molecular characterization

showed 25% of tumors classified as high-grade endometrioid have a molecular

phenotype similar to uterine serous carcinomas, including frequent TP53

mutations and extensive SCNA.

Ovarian Carcinoma

TCGA review for ovarian cancer included 489 clinically annotated stages II to IV

high-grade serous ovarian cancer (HGSC) samples and corresponding normal

DNA. The summary of mutational spectrum includes high prevalence of

mutations in TP53 (present in at least 96% of samples), BRCA1, and BRCA2

(including both germline and somatic mutations) in 22% of the tumors (76).

HGSC demonstrates a high frequency of mutations in genes involved in

homologous recombination, and is hence considered a disease of high genomic

instability. The mutation spectrum in HGSC differs from those in other histologic

subtypes of ovarian cancer. Clear-cell ovarian cancer tumors have few TP53

mutations, yet frequent ARID1A and PIK3CA mutations (39). Endometrioid

ovarian cancers carry frequent CTNNB1, ARID1A, and PIK3CA mutations and a

lower rate of TP53; and mucinous ovarian cancer tumors have prevalent KRAS

mutations (77). Such differences between ovarian cancer subtypes demonstrate

the ongoing need for subtype-specific targeted therapeutic modalities.

Cervical Carcinoma

TCGA study collection included primary frozen cervical cancer tumor tissue and

blood samples from patients who had not previously received chemotherapy or

radiotherapy. Whole exome sequencing demonstrated the following significantly

mutated genes: ERBB3, CASP8, HLA-A, SHKBP1, and TGFBR2 (78).

265Amplifications and fusion events were reported involving the BCAR4 gene, a

metastasis-promoting IncRNA that enhances cell proliferation in estrogenresistant breast cancer by activating the HER2/3 pathway, which can be targeted

indirectly by lapatinib (a dual tyrosine kinase inhibitor that interrupts the HER2

and EGFR pathways) (78,79). Amplifications in CD274 and PDCD1LG2 were

identified, both of which have been associated with response to lapatinib.

Another identified novel genomic characteristic that further subclassifies cervical

cancers is a group of endometrial-like cervical cancers involved predominantly

with HPV-negative tumors and characterized by mutations in KRAS, ARID1A,

and PTEN. These proteins may serve as therapeutic targets.

IMMUNOLOGY

The immune system plays an essential part in host defense mechanisms, in

particular the response to infections and neoplastic transformation. Our increased

understanding of immune system regulation provides opportunities for the

development of novel immunotherapeutic and immunodiagnostic approaches.

Immunologic Mechanisms

The human immune system has the potential to respond to abnormal or

tumor cells in various ways. Some of these immune responses occur in an

innate or antigen-nonspecific manner, whereas others are adaptive or

antigen specific. Adaptive responses are specific to a given antigen. The

establishment of a memory response allows a more rapid and vigorous response

to the same antigen in future encounters. Various innate and adaptive immune

mechanisms are involved in responses to tumors, including cytotoxicity directed

to tumor cells mediated by cytotoxic T cells (CTLs), natural killer (NK) cells,

macrophages, and antibody-dependent cytotoxicity mediated by complementation

activation (80).

Adaptive or specific immune responses include humoral and cellular

responses. Humoral immune responses refer to the production of antibodies.

Antibodies are bifunctional molecules composed of a variable region with

specific antigen-binding sites, combined with a constant region that directs the

biologic activities of the antibody, such as binding to phagocytic cells or

activation of complement. Cellular immune responses are antigen-specific

immune responses mediated directly by activated immune cells rather than

by the production of antibodies. The distinction between humoral and cellular

responses is historical and originates from the experimental observation that

humoral immune function can be transferred by serum, whereas cellular immune

function requires the transfer of cells. Most immune responses include both

266humoral and cellular components. Several types of cells, including cells from

both the myeloid and lymphoid lineages, make up the immune system. Specific

humoral and cellular immune responses to foreign antigens involve the

coordinated action of populations of lymphocytes operating in concert with one

another and with phagocytic cells (macrophages). These cellular interactions

include direct cognate interactions involving cell-to-cell contact and cellular

interactions involving the secretion of and response to cytokines or lymphokines.

Lymphoid cells are found in lymphoid tissues, such as lymph nodes or spleen, or

in the peripheral circulation. The cells that make up the immune system originate

from stem cells in the bone marrow.

B Cells, Hormonal Immunity, and Monoclonal Antibodies

B lymphocytes synthesize and secrete antibodies. Mature, antigen-responsive B

cells develop from pre-B cells (committed B-cell progenitors) and differentiate to

become plasma cells, which produce large quantities of antibodies. Pre-B cells

originate from bone marrow stem cells in adults after rearrangement of

immunoglobulin genes from their germ cell configuration. Mature B cells express

cell surface immunoglobulin molecules that function as receptors for antigen.

Upon interaction with antigen, mature B cells respond to become

antibody-producing cells. The process requires the presence of appropriate cell–

cell stimulatory signals and cytokines. Monoclonal antibodies are directed against

a specific antigenic determinant. In contrast, polyclonal antibodies detect multiple

epitopes that might be presented by just one or a panel of proteins. The in vitro

production of monoclonal antibodies, pioneered by Kohler and Milstein in the

1970s, has become an invaluable diagnostic and therapeutic tool, particularly for

the management of malignancies (81). The tumor antigen CA125, for example,

was detected in a screen of antibodies generated against ovarian cancer cell lines.

A radioimmunoassay is widely used to measure CA125 in the serum of patients

with ovarian cancer and guide treatment decisions. Therapeutic approaches

utilized immunotoxin-conjugated monoclonal antibodies directed to human

ovarian adenocarcinoma antigens. These antibodies induce tumor cell killing and

can prolong survival in mice implanted with a human ovarian cancer cell line.

However, some obstacles limit the clinical use of monoclonal antibodies,

including tumor cell antigenic heterogeneity, modulation of tumor-associated

antigens, and cross-reactivity of normal host and tumor-associated antigens.

Unique tumor-specific antigens have not been identified. All tumor antigens have

to be considered as tumor-related antigens because they are expressed on

malignant and nonmalignant tissues. Because most monoclonal antibodies are

murine, the host’s immune system can recognize and respond to these foreign

mouse proteins. The use of the genetically engineered monoclonal antibodies

267composed of human-constant regions with specific antigen-reactive murine

variable regions can result in reduced antigenicity.

T Lymphocytes and Cellular Immunity

[6] T lymphocytes have a central role in the generation of immune responses

by acting as helper cells in both humoral and cellular immune responses and

by acting as effector cells in cellular responses. T-cell precursors originate in

the bone marrow and move to the thymus, where they mature into functional T

cells. During their thymic maturation, T cells that can recognize antigens in the

context of the major histocompatibility complex (MHC) molecules are selected,

while self-responding T cells are removed (82).

T cells can be distinguished from other types of lymphocytes by their cell

surface phenotype, based on the pattern of expression of various molecules,

and by differences in their biologic functions. All mature T cells express certain

cell surface molecules, such as the cluster determinant 3 (CD3) molecular

complex and the T-cell antigen receptor (TCR), found in close association with

the CD3 complex. T cells recognize antigens through the cell surface T-cell

antigen receptor. The structure and organization of this molecule are similar to

those of antibody molecules, which are the B-cell receptors for the antigen.

During T-cell development, the T-cell receptor gene undergoes gene

arrangements similar to those seen in B cells, but there are important differences

between the antigen receptors on B cells and T cells. The T-cell receptor is not

secreted, and its structure is somewhat different from that of antibody molecules.

The way in which the B-cell and T-cell receptors interact with antigens is quite

different. T cells can respond to antigens only when these antigens are presented

in association with MHC molecules on antigen-presenting cells. Effective antigen

presentation involves the processing of the antigen into small fragments of

peptide within the antigen-presenting cell and the subsequent presentation of

these fragments of antigen in association with MHC molecules expressed on the

surface of the antigen-presenting cell. T cells can respond to antigens only when

presented in this manner, unlike B cells, which can bind antigens directly, without

processing and presentation by antigen-presenting cells.

[7] There are two major subsets of mature T cells that are phenotypically

and functionally distinct: T-helper/inducer cells, which express the CD4 cell

surface marker, and the CTLs, which express the CD8 marker. The

expression of these markers is acquired during the passage of T cells through the

thymus. CD4 T cells can provide help to B cells, resulting in the production of

antibodies by B cells, and interact with antigen presented by antigen-presenting

cells in association with MHC class II molecules. CD4 T cells can act as helper

cells for other T cells. CD8 T cells include cells that are cytotoxic (cells that can

268kill target cells bearing appropriate antigens), and they interact with an antigen

presented on target cells in association with MHC class I molecules. These T cells

can inhibit the biologic functions of B cells or other T cells (80). Although the

primary biologic role of CTLs seems to be lysis of virus-infected autologous cells,

cytotoxic immune T cells can mediate the lysis of tumor cells directly.

Presumably, CTLs recognize antigens associated with MHC class I molecules on

tumor cells through their antigen-specific T-cell receptor, setting off a series of

events that ultimately results in the lysis of the target cell.

Monocytes and Macrophages

Monocytes and macrophages, which are myeloid cells, have important roles in

innate and adaptive immune responses; macrophages play a key part in the

generation of immune responses. T cells do not respond to foreign antigens unless

those antigens are processed and presented by antigen-presenting cells.

Macrophages (and B cells and dendritic cells) express MHC class II

molecules and are effective antigen-presenting cells for CD4 T cells. Helperinducer (CD4) T cells that bear a T-cell receptor of appropriate antigen and selfspecificity are activated by this antigen-presenting cell to provide help (various

factors—lymphokines—that induce the activation of other lymphocytes). In

addition to their role as antigen-presenting cells, macrophages play an important

part in innate responses by ingesting and killing microorganisms. Activated

macrophages, besides their many other functional capabilities, can act as

cytotoxic, antitumor killer cells.

Natural Killer Cells

NK cells are effector cells in an innate type of immune response: the nonspecific

killing of tumor cells and virus-infected cells. Therefore, NK activity represents

an innate form of immunity that does not require an adaptive, memory

response for optimal biologic function, but the antitumor activity can be

increased by exposure to several agents, particularly cytokines such as

interleukin-2 (IL-2). Characteristically, NK cells have a large granular

lymphocyte morphology. NK cells display a pattern of cell surface markers that

differs from those characteristic of T or B cells. NK cells can express a receptor

for the crystallizable fragment (Fc) portion of antibodies, and other NK-associated

markers. NK cells appear to be functionally and phenotypically heterogeneous,

when compared with T or B cells. The cells that can carry out antibody-dependent

cellular cytotoxicity, or antibody-targeted cytotoxicity, are NK-like cells.

Antibody-dependent cellular cytotoxicity by NK-like cells resulted in the lysis of

tumor cells in vitro. The mechanisms of this tumor cell killing are not clearly

understood, although close cellular contact between the effector cell and the target

269cell seems to be required.

Cytokines, Lymphokines, and Immune Mediators

Many events in the generation of immune responses (and during the effector

phase of immune responses) require or are enhanced by cytokines, which are

soluble mediator molecules (Table 6-5) (83,84). Cytokines are pleiotropic in

that they have multiple biologic functions that depend on the type of target cell or

its maturational state. Cytokines are heterogeneous in the sense that most

cytokines share little structural or amino acid homology. Cytokines are called

monokines if they are derived from monocytes, lymphokines if they are

derived from lymphocytes, interleukins if they exert their actions on

leukocytes, or interferons (IFNs) if they have antiviral effects. They are

produced by a wide variety of cell types and seem to have important roles in

many biologic responses outside the immune response, such as inflammation or

hematopoiesis. They may be involved in the pathophysiology of a wide range of

diseases and show great potential as therapeutic agents in immunotherapy for

cancer. Although cytokines are a heterogeneous group of proteins, they share

some characteristics. For instance, most cytokines are low- to intermediatemolecular-weight (10 to 60 kDa) glycosylated-secreted proteins. They are

involved in immunity and inflammation, are produced transiently and locally

(they act in an autocrine and paracrine rather than an endocrine manner), are

extremely potent in small concentrations, and interact with high-affinity cellular

receptors that are specific for each cytokine. The cell surface binding of cytokines

by specific receptors results in signal transduction followed by changes in gene

expression and, ultimately, by changes in cellular proliferation or altered cell

behavior, or both. Their biologic actions overlap, and exposure of responsive cells

to multiple cytokines can result in synergistic or antagonistic biologic effects.

T-cell subsets characterized by the secretion of distinct patterns of cytokines

were identified. TH1 and TH2 are two helper T-cell subpopulations that

control the nature of an immune response by secreting a characteristic and

mutually antagonistic set of cytokines: clones of TH1 produce IL-2 and IFN-

γ, whereas TH2 clones produce IL-4, IL-5, and IL-10 (85). A similar

dichotomy between TH1- and TH2-type responses was reported in humans.

Human IL-10 inhibits the production of IFN-γ and other cytokines by human

peripheral blood mononuclear cells and by suppressing the release of cytokines

(IL-1, IL-6, IL-8, and TNF-α) by activated monocytes (84). IL-10 downregulates

class II MHC expression on monocytes, resulting in a strong reduction in the

antigen-presenting capacity of these cells. Together, these observations support

the concept that IL-10 has an important role as an immune-inhibitory cytokine.

270Additional T-cell subsets were identified, including Th17 cells and regulatory T

cells (Treg). Th17 cells are a distinct, pro-inflammatory T-cell subset, which is

functionally characterized by mediating protection against extracellular bacteria

and by its pathogenic role in autoimmune disorders (86,87). Th17 cells

characteristically produce IL-17, CXCL13 (a B-cell stimulatory chemokine), IL-

6, and TNF-α, in contrast to Th2 cells, which characteristically produce IL-4, IL-

5, IL-9, and IL-13, or Th1 cells, which produce IFN-γ (Fig. 6-2). Treg cells

constitute another subset of CD4-positive T cells that participates in the

maintenance of immunologic self-tolerance by actively suppressing the activation

and expansion of self-reactive lymphocytes. Treg cells are characterized by the

expression of CD25 (the IL-2 receptor chain) and the transcription factor FoxP3

(88,89). Treg cell activity is thought to be important in preventing the

development of autoimmune diseases. Removal of Treg may enhance immune

responses against infectious agents or cancer. Although much remains to be

learned about the role of Treg activity in antitumor immunity, it is clear that such

cells may play a role in modulating host responses to cancer. Much of the recent

research has been directed at the tumor microenvironment. Specifically, the tumor

microenvironment consists of several varieties of cell types that interact with

tumor cells to influence tumor initiation, growth, and metastasis (90). The tumor

microenvironment plays a key role in allowing tumor cells to evade immune

recognition and destruction.

Because epithelial cancers of the ovary usually remain confined to the

peritoneal cavity, even in the advanced stages of the disease, it was suggested

that the growth of ovarian cancer intraperitoneally could be related to a local

deficiency of antitumor immune effector mechanisms (91). Studies showed

that ascitic fluid from patients with ovarian cancer contained increased

concentrations of IL-10. Various other cytokines are seen in ascitic fluid obtained

from women with ovarian cancer, including IL-6, IL-10, TNF-α, granulocyte

colony-stimulating factor (G-CSF), and granulocyte-macrophage colonystimulating factor (GM-CSF). A similar pattern was seen in serum samples from

women with ovarian cancer with elevations of IL-6 and IL-10.

TNF-α is a cytokine that can be directly cytotoxic for tumor cells, increase

immune cell–mediated cellular cytotoxicity, activate macrophages, and

induce secretion of monokines. Other biologic activities of TNF-α include the

induction of cachexia, inflammation, and fever; it is an important mediator of

endotoxic shock.

Cytokines in Cancer Therapy

Cytokines are exceptionally pleiotropic with a wide array of biologic activities,

including some outside the immune system (92). Because some cytokines have

271direct or indirect antitumor and immune-enhancing effects, several of these

factors are used in the experimental treatment of cancer.

The precise roles of cytokines in antitumor responses have not been

elucidated. Cytokines can exert antitumor effects by many different direct or

indirect activities. It is possible that a single cytokine could increase tumor growth

directly by acting as a growth factor while at the same time increasing immune

responses directed toward the tumor. The potential of cytokines to increase

antitumor immune responses was tested in experimental adoptive immunotherapy

by exposing the patient’s peripheral blood cells or tumor-infiltrating lymphocytes

to cytokines such as IL-2 in vitro, thus generating activated cells with antitumor

effects that can be given back to the patient (93). Some cytokines can exert direct

antitumor effects. TNF can induce cell death in sensitive tumor cells.

The effects of cytokines on patients with cancer might be modulated by soluble

receptors or blocking factors. For instance, blocking factors for TNF and for

lymphotoxin were found in ascitic fluid from patients with ovarian cancer (93).

Such factors could inhibit the cytolytic effects of TNF or lymphotoxin and should

be taken into account in the design of clinical trials of intraperitoneal infusion of

these cytokines.

Table 6-5 Sources, Target Cells, and Biologic Activities of Cytokines Involved in

Immune Responses

272Cytokines have growth-increasing effects on tumor cells in addition to

inducing antitumor effects. They can act as autocrine or paracrine growth

factors for human tumor cells, including those of nonlymphoid origin. For

instance, IL-6 (which is produced by various types of human tumor cells) can act

as a growth factor for human myeloma, Kaposi sarcoma, renal carcinoma, and

epithelial ovarian cancer cells (94).

273Cytokines are of great potential value in the treatment of cancer, but because of

their multiple, even conflicting, biologic effects, a thorough understanding of

cytokine biology is essential for their successful use (93).

FACTORS THAT TRIGGER NEOPLASIA

Cell biology is characterized by considerable redundancy and functional overlap,

so a defect in one mechanism does not invariably jeopardize the function of the

cell. When a sufficient number of abnormalities in structure and function occur,

normal cell activity is jeopardized, and uncontrolled cell growth or cell death

results. Either end point may result from accumulated genetic mutations. Factors

are identified that enhance the likelihood of genetic mutations, jeopardize normal

cell biology, and may increase the risk of cancer.

Increased Age

Increasing age is considered the single most important risk factor for the

development of cancer (95). Cancer is diagnosed in as much as 50% of the

population by 75 years of age. It was suggested that the increasing risk of cancer

with age reflects the accumulation of critical genetic mutations over time, which

ultimately culminates in neoplastic transformation. The basic premise of the

multistep somatic mutation theory of carcinogenesis is that genetic or epigenetic

alterations of numerous independent genes result in cancer. Factors that are

associated with an increased likelihood of cancer include exposure to exogenous

mutagens, altered host immune function, and certain inherited genetic syndromes

and disorders.

Environmental Factors

A mutagen is a compound that results in a genetic mutation. A number of

environmental pollutants act as mutagens when tested in vitro. Environmental

mutagens usually produce specific types of mutations that can be differentiated

from spontaneous mutations. A carcinogen is a compound that can produce

cancer. It is important to recognize that all carcinogens are not mutagens and that

all mutagens are not necessarily carcinogens.

Smoking

Cigarette smoking is perhaps the most well-known example of mutagen

exposure that is associated with the development of lung cancer when the

exposure is of sufficient duration and quantity in a susceptible individual. An

association between cigarette smoking and cervical cancer has been recognized

274for decades. It was determined that the mutagens in cigarette smoke are

selectively concentrated in cervical mucus (96). It was hypothesized that

cigarette smoke acted as a mutagen when exposed to the proliferating epithelial

cells of the transformation zone, thus increasing the likelihood of DNA damage

and subsequent cellular transformation.

Others observed that HPV DNA is frequently inserted into the fragile

histidine triad (FHIT) gene in cervical cancer specimens. The FHIT is an

important tumor suppressor gene. Cigarette smoking might facilitate the

incorporation of HPV DNA into the FHIT gene with subsequent disruption

of correct tumor suppressor gene function.

Radiation

Radiation exposure can increase the risk of cancer. The biologic effects of

ionization surpass those of x-rays and gamma rays. Ionization of water molecules

creates hydroxyl radicals, which cause DNA strand breaks or base damage. Most

radiation-induced damage is repaired precipitously. However, misrepair may

cause point mutations, chromosome translocations, and gene fusions associated

with cancer initiation (97). Normally, radiation damage prompts an S-phase arrest

so that DNA damage is repaired. This requires normal p53 gene function. If DNA

repair fails, the damaged DNA is propagated to daughter cells following mitosis.

If a sufficient number of critical genes are mutated, cellular transformation may

result (98).

Immune Function

Systemic immune dysfunction was recognized for decades as a risk factor for

cancer. Patients infected with HIV who have a depressed CD4 cell count are

reported to be at increased risk of cervical dysplasia (99). Individuals who

underwent high-dose chemotherapy with stem cell support may be at increased

risk of developing a variety of solid neoplasms. These examples illustrate the

importance of immune function in host surveillance for transformed cells.

Another example of altered immune function that may be related to the

development of cervical dysplasia is the alteration in mucosal immune function

that occurs in women who smoke cigarettes (100). The Langerhans cell

population of the cervix is decreased in women who smoke. Langerhans cells are

responsible for antigen processing. It is postulated that a reduction in these cells

increases the likelihood of successful HPV infection of the cervix.



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