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2004 Fetal blood group genotyping from DNA from maternal plasma: an important advance in the management and prevention of haemolytic disease of the fetus and newborn.. Haematologica 53Su

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contained mixtures of several antibodies This problemwas partly overcome by applying extensive statisticalanalysis to the results (van Rood and van Leeuwen1963) in order to define single specificities Progressbecame more rapid with (1) the replacement of leuco-agglutination by lymphocytotoxicity tests and (2) therealization that HLA antibodies are frequently formed

in pregnancy, particularly as these antibodies, in trast with those formed after blood transfusion aredirected against a limited number of HLA antigens(Payne and Rolfs 1958; van Rood 1958) and (3) theorganization of international histocompatibility work-shops in which different laboratories were able to com-pare results by sharing reagents and typing commonpanels of cells These workshops, which continue on aregular basis, have been instrumental in the orderlydevelopment of the HLA system and its nomenclature(Bodmer 1997) The HLA antigens first detected werefound to be encoded by three closely linked genes:

con-HLA-A, -B and C, subsequently named class I genes.

The observation that lymphocytes from two related individuals can stimulate each other to blast formation when cultured together (mixed lymphocyteculture (MLC) assay), and that the antigens respons-ible for this stimulation are inherited together withHLA antigens, led to the discovery of HLA-D antigens

un-(Bach and Hirschhorn 1964; Bain et al 1964), which

were later detected serologically on B cells and named

DR antigens (van Rood et al 1975, 1976; van Rood

and van Leeuwen 1976) HLA-DR molecules togetherwith HLA-DQ and HLA-DP constitute the classicalclass II molecules

The ability to study HLA genes and their alleles

at the molecular level has enormously advanced the

and plasma components

13

Antigens expressed on leucocytes and platelets include

HLA class I and II molecules as well as those that are

specific for particular cells and those that are shared

with red cells In this chapter, these antigens, their

structure, function and corresponding antibodies

are described, together with methods for detecting

them The chapter includes an account of the clinical

relevance of these systems and effects owing to related

incompatibilities

The human leucocyte antigen (HLA)

system

The human leucocyte antigens (HLAs), coded by genes

of the major histocompatibility complex (MHC) are

cell surface glycoproteins that are critical in

deter-mining the compatibility of tissue grafts, in selecting

donor–recipient pairs for transfusion and

transplanta-tion and in designing epitope-specific cellular

immuno-therapy HLA antibodies are commonly formed after

blood transfusion and pregnancy The name is

mis-leading HLA molecules are expressed on a wide range

of cells in addition to leucocytes, and although these

molecules prove ‘alloantigenic’ to gravid women and

to transfusion and graft recipients, they function not

as antigens but as peptide chaperones crucial to the

process of adaptive immunity (Paul 2003; Wang et al.

2005) HLA molecules play a key role in host defences

by presenting foreign antigens to the immune

The first HLA antigen to be clearly defined was

HLA-A2, first named MAC (Dausset 1958) Early

work on the definition of HLA antigens was

complic-ated by the poor reproducibility of the

leucoagglutina-tion assay and by the fact that the early seroreagents

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knowledge of the HLA genes Not only is the region

now known ‘nucleotide by nucleotide’ at the genome

level, but also hundreds of alleles of the different loci

have been sequenced in the population Most HLA

typing is now done at the DNA level

Human leucocyte antigen: the human major

histocompatibility complex

The name MHC refers to the ability of the genes of this

genomic region to determine graft rejection between

individuals of the same species In the 1960s, the HLA

genes, first discovered through leucocyte

agglutina-tion, were established as the genes responsible for graft

rejection in man The physiologic function of these

molecules was determined during the following decade:

presentation of antigens to T cells (Zinkernagel and

Doherty 1974) The mechanism of this ‘HLA

restric-tion’ was first explained by Townsend and co-workers

(1986), who showed that synthetic peptides could be

presented to the T cell The final explanation of how

this peptide could be presented to the T cell awaited

the discovery of the structure of the HLA molecules in

1987 and the crystallization of a T-cell receptor bound

to an MHC molecule

The extremely polymorphic, closely linked genes of

the HLA system are located in a region that spans

about 4000 kilobases (kb) on the short arm of

chromo-some 6 (Breuning et al 1977) Moving from

centro-mere to telocentro-mere, the class II genes are separated

from the class I genes by a number of functionally

unrelated genes (and pseudogenes) that encode the

complement factors C2, C4a and C4b heat shock

proteins, cytokines and enzymes (class III genes)

(Fig 13.1)

After three decades of maps of ever increasing

elaboration, the complete sequence of the human

MHC was published in 1999 by the MHC Sequencing

Consortium (1999)

Class I region

HLA-A, -B and -C code for the heavy chain of the

MHC class I molecules expressed on most cells

HLA-F, -G and -E code for the heavy chain of non-classical

class I molecules, with highly specialized functions

The MIC genes or human MHC class I chain-related

genes encode stress-inducible proteins implicated in

the regulation of NK cell activity HFE is a class I-like

gene located approximately 4 Mb telomeric of HLA-Fand responsible for most hereditary haemochromatosis

Class III region

TNFB and -A code for tumour necrosis factors, HSPgenes for heat shock proteins, C2, Bf and C genes forproteins of the complement system, and P450-C21Bfor a steroid 21-hydroxylase

Class II region

The HLA-DRBA1, -DQA1 and -DPA1 genes code foralpha chains of the DR, DQ and DP class II molecules.HLA-DRB1, -DQB1 and -DPB1 code for the betachain of the DR, DQ and DP class II molecules Inaddition to HLA-DRB1, which codes for the primaryHLA specificities such as DR1, DR2, DR4, etc., otherDRB genes code for the beta chain of the specificitiesDR52 (DRB3), DR53 (DRB4) and DR51 (DRB5) notpresent in all haplotypes The DO and DM mole-cules regulate the loading of exogenous peptides intoclass II molecules LMP2 and LMP7, which encode the subunits of the proteasome, and TAP1 and TAP2,which encode a peptide transporter, are involved in the processing and presentation of antigens by class Imolecules

HLA class I and II molecules: structure and function

HLA molecules engage two distinct arms of the mediated immune response MHC class I moleculespresent antigen to cytotoxic T cells (CTLs), whereasMHC class II present to helper T cells Antigens are notpresented by HLA molecules as intact proteins Theantigen must first be degraded to peptide fragmentsand presented in the context of the HLA molecule tothe T-cell receptor

T-cell-HLA class I molecules

HLA-A, -B and -C genes produce a transmembraneglycosylated polypeptide of molecular weight 4300(the α or heavy chain) linked non-covalently to β2-microglobulin, a non-polymorphic and non-glycosylated polypeptide of molecular weight 1200(the β or light chain), which is encoded by a gene on

chromosome 15 (Snary et al 1977a; Barnstable et al.

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100- 200- 300- 400- 500- 600- 700- 800- 900- 1000- 1100- 1200- 1300- 1400- 1500- 1600- 1700- 1800- 1900- 2000- 2100- 2200- 2300- 2400- 2500- 2600- 2700- 2800- 2900- 3000- 3100- 3200- 3300- 3400-

0-HFE

C L A S S I

C L A S S I I I

C L A S S I I

HLA-F MICE

HLA-DPA2 HLA-DPA3 HLA-DPB2

HLA-A

HLA-E

HLA-C HLA-B

MICA

TNFA

HSPA1L

HLA-DOB TAP2 LMP7 TAP1 LMP2 HLA-DMB HLA-DMA HLA-DOA

HSPA1A HSPA1B C2 C4B P450-C21B

HLA-DRA HLA-DRB3 HLA-DRB1 HLA-DQA1 HLA-DQB1

HLA-DPA1 HLA-DPB1

BF

MICB TNFB

Fig 13.1 The segment of the small arm of chromosome 6

that contains the HLA complex is shown in detail The first

bar shows the division of the complex into class I, II and III

regions The ruler indicates the number of kilobases The

genomic map shows the approximate positions of the gene

loci mentioned in the text Bars to the right show expressed

genes and to the left, pseudogenes (not expressed) In bold

are shown the genes coding for the heavy chain of the classical class I molecules and the alpha and beta chains of

the class II molecules Class I region: HLA-A, B and C code

for the heavy chain of the MHC class I molecules expressed

on most cells HLA-F, G and E code for the heavy chain of non-classical class I molecules, with highly specialized functions The MIC genes or human MHC class I

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1978) The extracellular part of the heavy chain

con-sists of three domains: α1, α2 and α3 (Fig 13.2)

The three-dimensional structure of class I molecules

has been revealed by X-ray crystallographic analysis,

first of A2 (Fig 13.3) and subsequently of

HLA-A68 and HLA-B27 (Bjorkman et al 1987; Garretti

et al 1989; Madden et al 1991) The α3 and β2m

domains have tertiary structures similar to domains in

the constant region of the immunoglobulins The top

of the molecule is formed by pairing the α1 and α3

domains, which together form the antigen

peptide-binding groove The majority of the polymorphic

determinants in class I molecules occur on the floor of

this cleft (Bjorkman et al 1987) (see Fig 13.3) The

class I molecules specifically bind peptides of defined

length, usually 6–10 residues (Falk et al 1991) All

peptides bind similarly with their N- and C-terminisequestered in the binding groove by a network ofhydrogen bonds to residues conserved in all class I glycoproteins (Madden and Wiley 1992) In addition,there are allele-specific binding pockets with a strongpreference for a few side-chains at some positions ofthe peptide This explains the correlation betweenclass I polymorphism and the affinity of peptide bind-

ing (Falk et al 1991) Generally, these peptides derive

from self-proteins, but in virus-infected cells the tides from the pathogen may be processed in thecytosol and migrate with the HLA molecules to the cellsurface (Pamer and Cresswell 1998) Tumour antigenscan be detected in the same way Class I molecules are expressed on most cells and they inform the scan-ning CTL about the status of potential target cells for

pep-Fig 13.1 (cont.)

chain-related genes encode stress-inducible proteins

implicated in the regulation of NK cell activity HFE is a

class I-like gene located approximately 4 Mb telomeric of

HLA-F and responsible for hereditary haemochromatosis.

Class III region: TNFB and A code for tumour necrosis

factors, HSP genes for heat shock proteins, C2, Bf and

C genes for proteins of the complement system, and

P450-C21B for a steroid 21-hydroxylase Class II region:

The HLA-DRBA1, -DQA1 and -DPA1 genes code for

alpha chains of the DR, DQ and DP class II molecules

HLA-DRB1, -DQB1 and -DPB1 code for the beta chain of the DR, DQ and DP class II molecules In addition to HLA- DRB1, which codes for the primary HLA specificities such as DR1, DR2, DR4, etc., other DRB genes code for the beta chain of the specificities DR52 (DRB3), DR53 (DRB4) and DR51 (DRB5) not present in all haplotypes The DO and

DM molecules regulate the loading of exogenous peptides into class II molecules LMP2 and LMP7, which encode the subunits of the proteasome, and TAP1 and TAP2, which, encode a peptide transporter, are involved in the processing and presentation of antigens by class I molecules.

Fig 13.2 Structure of class I and class

II HLA molecules showing domains

and transmembrane segments From

Roitt and Delves (2001).

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destruction Peptide epitopes presented by them can

only be recognized by CTL if these (1) have a specific

receptor for the antigen and (2) the same HLA class

antigens as the target cell This phenomenon, known

as HLA restriction, was first described in the mouse

(Zinkernagel and Doherty 1974) The HLA-A, -B and

-C antigens are expressed on all nucleated cells except

spermatozoa and placental trophoblast The antigens

are also found on platelets and some class I antigens

have been detected on red cells The number of class I

molecules on various cells differs and, particularly on

platelets, some of the antigens are weakly expressed

HLA class II molecules

All typical class II molecules consist of two

transmem-brane glycoprotein chains of molecular weight 33 kDa

(the heavy or α-chain) and 28 kDa (the light or

β-chain) respectively (Snary et al 1977b) The

extracel-lular component of both chains consists of two distinctdomains: α1, α2 and β1, β2 The domains distal to thecell surface carry most of the polymorphic determinants.The constant domain near the cell surface is very similar

to the constant domain of the immunoglobulin heavy

chains (Shackelford et al 1982; see also Fig 13.2).

The crystal structure of class II molecules (DR1) issimilar to that of class I molecules; polymorphic deter-minants of class II molecules are also clustered in the

antigen peptide-binding groove (Brown et al 1993) In

contrast with class I molecules, class II molecules bindlonger peptides with no apparent restriction on pep-

tide length (Rudensky et al 1991) The peptides bind

to the groove as a straight extended chain with a singlepronounced twist Hydrogen bonds along the main

(b)

(a)

Peptide-binding cleft

N N

Fig 13.3 Schematic representations of the crystallized

structure of the HLA-A2 molecule (a) The four domains,

with the α 1 and α 2 domains forming a putative

peptide-binding region (b) Top surface of the molecule The putative antigen-binding groove is shown, made up of a β-pleated sheet flanked by two α-helices (Bjorkman et al 1987).

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chain of all peptides interact with residues from the

α-helical regions and the β-sheet in the peptide-binding

groove and thus provide a binding component that

is independent of the sequence of the peptide Twelve

hydrogen bonds on the peptide bind to determinants

encoded by residues conserved in most class II alleles

and this suggests that peptides bind to class II

molecules by a universal mode However, particular

side-chains of the peptide are accommodated in

poly-morphic pockets in the binding groove that determine

specific binding of peptides and thus the affinity of

the peptide class II molecule bond (Stern et al 1994).

The expression of class II antigen is restricted to B cells

and to antigen-presenting cells such as macrophages,

dendritic cells and Langerhans cells Class II antigens

are also present on activated T lymphocytes and some

tumour cells (Winchester and Kunkel 1979)

The cells that express class II molecules, specialized

antigen-presenting cells (APCs), such as dendritic cells,

mononuclear phagocytes and B cells, bind

exogen-ously derived peptides of 9–22 residues In the case of

macrophages and B cells, the HLA molecule–antigen

complex is assembled within intracellular organelles

With all APCs the antigen peptides are held in a groove

in HLA class II molecules, and this plasma

membrane-bound compound antigen is recognized by helper T

lymphocytes via their T-cell receptors during

immuno-surveillance The polymorphic HLA determinants in

the peptide-binding groove of the class II molecule

strongly influence peptide binding

Dendritic cells, which express HLA class II antigens

particularly well, are the APCs that present antigen to

helper T cells to induce a primary immune response

Memory T cells can be stimulated by macrophages, B

cells and even by free antigens (Berg et al 1994) Class

II antigen complexes instruct the helper T-cell system

to initiate the humoral immune response and assist

in the cellular immune process; for this reason, class II

genes are often referred to as ‘immune response genes’

HLA genes and antigens

The linkage between the HLA genes is so strong that

crossing over between them is rare; therefore the alleles

of the HLA genes present on one chromosome usually

segregate together within a family The two alleles of

each individual gene are expressed co-dominantly (e.g

HLA-A1, -A11) The set of HLA alleles present on a

single chromosome is known as a haplotype Siblings

who inherit the same haplotypes from their parents are thus HLA identical, unless crossing over betweenHLA genes has occurred

As crossing over within the HLA region is rare, withrandom assortment equilibrium should be reached in apopulation over a long period of time; particular com-

binations of alleles at, for example, the A and B loci

or at the loci of the D region should not be more

com-mon than predicted from the product of their relative frequencies in the population However, in any givenpopulation, certain combinations of alleles or haplo-types are more frequent than expected, a phenomenonknown as ‘linkage disequilibrium’ For example, thefrequency of HLA-A1-B8 in European white people is8.8%, whereas the expected frequency of this haplo-type, based on the individual frequencies of A1 and B8, is 1.6% Selective pressures that affect survival orreproductive capacity usually drive linkage disequilib-rium Patterns of linkage disequilibrium vary in differ-ent populations

Nomenclature

A history of the development of HLA system nomenclature has been compiled by Boolmer (1997).Nomenclature for the HLA genes and antigens is regu-larly updated by the WHO Nomenclature Committeefor factors of the HLA system (Marsh 2003) For thenon-aficionado, nomenclature remains a challenge,because two systems remain in general use The olderserological nomenclature relies on identification ofantigens on the leucocyte surface (HLA antigens)

(Tiercy et al 2002) The following terms are used

for the ‘classical’ HLA antigens: for class I, the capital

letters -A, -B -C are appended to identify the locus; for class II, the prefix D followed by a letter (-DR, -DQ, -DP) for the subregion and by the letters A or B

to indicate whether the gene codes for the α- or chain, for example DRA, DQB, etc The letters are followed by a number that identifies epitopes deter-mined by alloantibodies or less often alloreactive cytotoxic T cells

β-Sequence-based nomenclature separates the HLAlocus with an asterisk (*) followed by four digits used

to designate the alleles of a particular gene; the firsttwo digits describe the serologically defined antigenwith which the allele is (or alleles are) most closelyassociated, and the last two or three digits completethe number of the allele as defined by molecular

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techniques (DNA typing, oligonucleotide typing,

nucleotide and amino acid sequencing, cloning), for

example HLA-A*0101 for the allele that encodes the

A1 antigen and A*0201, A*0202, etc for the alleles

associated with the antigen A2; the serologically

defined antigens encoded by alleles of each gene are

also numbered: A1, A2 etc.; w (‘workshop’) was used

to indicate that the specificity was provisional, but

in the future all serological specificities will be named

on the basis of correlation with an identified sequence

The letter ‘w’ can therefore be dropped with three sets

of exceptions: (1) Bw4 and Bw6 to distinguish them

as epitopes from those encoded by other alleles of

the HLA-B gene; (2) the C antigens for which the w

is retained throughout to avoid confusion with the

nomenclature of the complement system; and (3) the

Dw specificities defined by the MLC assay; and the

DP specificities defined by a secondary response of T

lymphocytes that had been primed by a first step in

the MLC (primed lymphocyte typing) (Bodmer et al.

1992)

One of the non-classical HLA class I genes, the

HLA-G gene, encodes a non-polymorphic α-chain

with a shortened cytoplasmic segment The HLA-G

molecule is expressed only on the trophoblast, which

suggests that it may have a role in embryonic

develop-ment or fetal–maternal immune interactions, or both

(Geraghty et al 1987; Kovats et al 1991) Class I

anti-gens are detected in a lymphocytotoxicity test, using

either alloantibodies or human or murine monoclonal

antibodies

Some 250 alleles of the class I A gene, about twice

this number of B alleles and 119 C alleles are

recog-nized by the WHO Nomenclature Committee The

numbers have been increasing rapidly For a list of

these and of class I antigens, see Marsh (2003)

Class II genes and antigens

The polymorphism of the class II genes is much greater

than detected on their products by serological typing

and by the MLC assay Studies at the DNA level have

shown that, in addition to the classical DR, DQ and

DP series of genes, there are several other non-classical

class II genes in the D region: DOA, DOB, DNA and

the DMA and DMB genes In addition, in the class II

chromosomal region, there are four genes, TAP1 and

TAP2, and LMP2 and LMP7, which encode molecules

involved in antigen processing (see below)

In the DR subregion there is a single α-chain gene

(DRA) with two alleles that, however, do not encode a

polymorphism on the α-chain There are nine DRB genes, five of which are pseudogenes (DRB2, DRB6, DRB7, DRB8 and DRB9) Seven of the DRB genes (except DRB1 and DRB9) are restricted to certain

DR haplotypes The genomic organization of the DR

region is shown in Figure 13.4 The DRB1, DRB3, DRB4 and DRB5 genes encode four separate β-

chains DRB1 encodes the major DR antigens, whereas the DRB3 gene codes for DR52, and the DRB5 gene for DR51.

In the DQ subregion there are two α genes: DQA1,

which encodes an α-chain and DQA2, which is not known to be expressed There are three B genes: DQB1, which encodes a β-chain and DQB2 and DQB3, not known to be expressed DQA1 and DQB1 are polymorphic but only the products of the DQB1

alleles have been serologically recognized (DQ antigens)

In the DP subregion there are two α and two β genes:

DPA1, DPA2, DPB1 and DPB2 DPA1 and DPB1

encode an α- and a β-chain respectively, whereas DPA2 and DPB2 are pseudogenes.

Additional HLA class II genes

The genes DOB and DNA located between the DP and

DQ subregions encode a β- and α-chain, respectively,

whose function is as yet unknown The genes DMA and DMB encode an α- and a β-chain, which associate

to form a class II molecule that contains a binding groove involved in antigen presentation (Kelly

peptide-et al 1991) The DM genes are polymorphic, but the

polymorphism is limited and the resulting antigenicdeterminants occur only on the area of the extracellu-lar portion of the protein that is proximal to the cellmembrane They therefore do not occur in the peptide-binding groove and thus do not affect antigen pre-sentation, in which the DM molecule probably has a

specialized function (Sanderson et al 1994).

Non-HLA genes involved in antigen processing

The TAP1 and TAP2 genes, located between DBO and DNA, do not encode typical class II proteins

but instead encode an important peptide transportermolecule involved in the endogenous processing of

antigen (Spies et al 1991) The TAP genes are morphic (Colonna et al 1992; Powis et al 1992a,b,

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poly-1993) This polymorphism may affect antigen

process-ing and thus the immune response, but at present this

is speculation The LMP2 and LMP7 genes, located

near the TAP genes, encode proteasomes that affect

the degradation of antigen molecules to peptides

(Cerundolo et al 1995).

The class II antigens are detected by alloantibodies

and, in some cases, also by monoclonal antibodies, using

a complement-dependent cytotoxicity test on isolated

B lymphocytes or a two-colour immunofluorescence

test on unseparated cells (van Rood et al 1976).

As mentioned above, a polymorphism (Dw) encoded

by the D region has been defined by using homozygous

typing cells in the mixed lymphocyte culture (MLC)

The exact relationship between Dw determinants and

the polymorphic determinants encoded by the DR, DP

genes is not known However, a strong correlation has

been observed between matching and mismatching for

DR polymorphic DNA sequences and reactivity in the

MLC The lymphocytes of all DR mismatched pairs

were reactive and 37% of the matched pairs were

non-reactive in the MLC The MLC reactivity of 63% of

the matched pairs may be due to unrecognized DR

alleles (Baxter-Lowe et al 1992), but incompatibility

for HLA-DP specificities has also been found to inducesignificant proliferation in the primary MLC in HLA-

A, -B, -DR and -DQ identical subjects (Olerup et al 1990) HLA-DQA1 or -DQB1 allele differences are not

important in the primary MLC among otherwise HLA

identical, unrelated subjects (Termijtelen et al 1991).

For a list of the very numerous class II alleles, antigens and determinants agreed by the WHONomenclature Committee, see Marsh (2003)

Other genes in the HLA region

As the products of these genes are not leucocyte gens and are not involved in antigen presentation byHLA molecules, they will not be discussed further

anti-Crossreactions in the HLA system

Sera from subjects alloimmunized against HLA gens are frequently crossreactive, as shown by the following example: a given serum may react with twodifferent serologically defined antigens, for example

Fig 13.4 Genomic organization of the HLA-DR region and

encoded products (specificities) (Bodmer et al 1992).

Pseudogenes are indicated by shaded boxes, expressed genes

by open boxes The serological specificity encoded by a gene

is shown underneath in italics *, rarely observed haplotypes;

#, DR51 and DR53 may not be expressed on certain haplotypes; §, the presence of DRB9 in these haplotypes needs confirmation.

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HLA-B51 and HLA-B52, but antibodies recognizing

these two specificities individually cannot be separated

from the serum The antibodies in such a serum are

in fact directed against a different antigen, HLA-B5

in this example, which occurs together with B51 and

B52 Thus the antibodies (anti-B5) crossreact with

B51- and B52-positive cells

This kind of crossreactivity is explained by the

mul-tiple mutations within the HLA genes A single allele of

a gene may code for different, separate polymorphisms

on the single HLA molecule it produces The

frequen-cies of the epitopes encoded by these different

poly-morphisms within a single allele differ greatly Some

have a very high frequency, for example HLA-B4 and

HLA-B6, and are called supratypic or ‘public’

anti-gens At the other end of the scale are antigens with a

very low frequency (1–2%) that are called ‘private’

antigens Thus, epitopes with different frequencies in

the population, and against which separate

alloanti-bodies can be made, occur on a single HLA molecule

and are the basis of the crossreactivity: antibodies

against public antigens (also called crossreactive

gens) react with cells carrying different private

anti-gens Particular public antigens occur together with

particular private antigens that are said to form

cross-reactive groups (CREGs) of (private) antigens The

higher the frequency of the public antigen in the

popu-lation, the more important are antibodies against the

antigen for crossreactivity Thus, anti-HLA-B4 and

-B6 are responsible for much of the crossreactivity

among the HLA-B antigens

The occurrence of crossreactive antigens is also

responsible for what are called ‘splits’ of HLA

anti-gens Frequently, a crossreactive antigen, for example

the antigen B5 in the above example, was defined

before the two private antigens B51 and B52, together

with which it occurs Later, when antibodies

recogniz-ing B51 and B52 were found, the B5 antigen was ‘split’

into B51 and B52 (see Table 13.2)

Soluble HLA class I antigens in plasma

Using monoclonal antibodies coated on to

immuno-beads and one-dimensional isoelectric focusing,

fol-lowed by immunoblotting using specific class I antisera,

all antigens defined to date have been detected in

plasma (Doxiadis and Grosse-Wilde 1989)

Soluble HLA class I antigens (sHLA) may have

important immunological effects sHLA have been

shown to inhibit alloreactive cytotoxic T cells

(Zavazava et al 1991) and to block specifically the

induction of HLA alloantibody formation (Grumet

et al 1994) Allogeneic sHLA alone or complexed with

antibody induces prolongation of allograft survival

(Sumimoto and Kamada 1990; Wang et al 1993a).

When a graft is rejected, sHLA is shed from it and thus graft rejection episodes can be identified by serial

measurements of donor-specific sHLA (Claas et al 1993; Puppo et al 1994).

HLA antibodies

Mechanisms of alloimmunization

The high density of HLA molecules on the cell surfacerenders allogeneic leucocytes highly immunogenic fol-lowing transfusion or pregnancy Sensitization dependsupon both donor and recipient factors Two recipientT-cell recognition mechanisms have been shown to becritical for the initiation of alloimmunity (Semple andFreedman 2002) The direct pathway occurs whenrecipient helper T cells interact with MHC molecules

on donor APCs The indirect pathway is more ous to the normal immune response Indirect recogni-tion involves processing of allogeneic donor molecules

analog-by recipient APCs and presentation to recipient helper

T cells With indirect allorecognition, interactions tween donor antigen and recipient APCs are essentialfor T-cell activation and subsequent antibody forma-tion For both pathways, the MHC molecules areexpressed on the surface of the APC and are availablefor presentation to circulating T cells If a T cell has areceptor with sufficient affinity for the peptide–MHCcombination (first signal) and various co-stimulatory(second signal) events occur, the T cell will be activatedand differentiate into an effector cell Cytokines such

be-as interleukin 2 (IL-2), IL-4 and alpha-interferon α) secreted from the activated helper cells stimulatedonor MHC class I-primed B cells to differentiate intothe plasma cells that secrete IgG antibodies and helper

(IFN-T cells (Weiss and Samuelsen 2003)

Development of HLA antibodies after transfusion

Unless measures are taken to reduce the number oftransfused leucocytes (see below), a high incidence ofHLA antibodies will be encountered in patients who

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receive multiple transfusions from different donors.

However, even in subjects exposed to the blood of a

single donor, the incidence of HLA antibodies is high

In a series in which patients awaiting renal grafting

were given three transfusions at 2-weekly intervals

from a potential donor, who in each case had a

haplo-type identical with one of the recipient’s haplohaplo-types,

HLA antibodies developed in some 30% of recipients

(Salvatierra et al 1980).

After the massive blood transfusion that used to be

associated with open heart surgery, lymphocytotoxic

antibodies and/or leucoagglutinins could be found in

almost all subjects, provided that repeated tests are

made, as some of the antibodies can be detected only

transiently In a series in which patients were tested at

1 week and usually also at 2, 4 and 12 weeks after open

heart surgery, 52 out of 54 developed leucocyte

anti-bodies; 12 weeks after transfusion antibodies were

present in only 62.5% of the subjects (Gleichmann

and Breininger 1975) The majority of HLA antibodies

formed after blood transfusion are directed against

class I antigens HLA antibodies are the most

import-ant cause of import-antibody-mediated refractoriness to

platelet transfusions (see later), of febrile transfusion

reactions prior to leucoreduction and of

transfusion-associated acute lung injury (see Chapter 15)

Some patients never become immunized despite

repeated transfusions of blood or of platelets Such

subjects are considered to be non-responders to HLA

Development of HLA antibodies in pregnancy

In primiparous women, lymphocytotoxic class I

anti-bodies may be found as early as the twenty-fourth

week of pregnancy and are present by the last trimester

in 10% of women (Overweg and Engelfriet 1969)

Estimates of the incidence of lymphocytotoxic

anti-bodies after a first pregnancy vary widely: 4.3%

(Ahrons 1971), 9.1% (Nymand 1974), 13% (Overweg

and Engelfriet 1969) and 25% (Goodman and Masaitis

1967) The discrepancies may well be due to the

vary-ing extent of the panels of lymphocytes with which the

sera were tested and the sensitivity of the techniques

applied The majority of HLA antibodies developed in

pregnancy are directed against class I antigens

Women tend to make antibodies against only

cer-tain of the HLA antigens to which they are exposed

during pregnancy In multiparous women who had

had at least four pregnancies, and were therefore likely

to have been exposed to antigens encoded by both oftheir partner’s haplotypes, the frequency of womenwith antibodies against only a single paternal antigenwas the same as that in primiparous women (Tongio

et al 1985) Both maternal and paternal (fetal) HLA

antigens play a role in the class I differential

immuno-genicity (Dankers et al 2003).

Although HLA antibodies are usually IgG, they produce no obvious damage to the fetus, presumablybecause they are absorbed by fetal cells in the placenta

Monoclonal HLA antibodies

Most murine monoclonal HLA antibodies are directedagainst non-polymorphic determinants of the HLA

molecules (Brodsky et al 1979; Trucco et al 1980;

1979); some antibodies detect a polymorphism that isdifferent from those detected by alloantisera (Quaranta1980) However, many murine monoclonals whichrecognize HLA antigens as defined by alloantiserahave been described, particularly anti-DR and anti-

DQ (Marsh and Bodmer 1989)

In addition, many human monoclonal HLA bodies against both class I and class II antigens havenow been described

anti-Some features of HLA antibodies

HLA antibodies formed after blood transfusion orpregnancy are characteristically IgG They are comple-ment activating and have cytotoxic properties and,like most granulocyte-reactive IgG antibodies, are leucoagglutinins (see below) HLA antibodies may benaturally occurring Using very sensitive techniques,weak HLA antibodies, particularly anti-B8, have beendemonstrated in the serum of about 1% of normaldonors who had had no known pregnancies or trans-

fusions (Tongio et al 1985) These antibodies are

usu-ally IgM and, in the cytotoxicity test, react only with

B cells, on which class I antigens are more stronglyexpressed than on T cells

HLA and haematopoietic progenitor cell grafts

Graft-versus-host-disease and graft-versus-tumour or -leukaemia

Haematopoietic cell (marrow, umbilical cord blood

or mobilized peripheral blood) transplantation is

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performed to replace inadequate or defective blood

cell production, for example in aplastic anaemia, sickle

cell disease and thalassaemia (Walters et al 2000; La

Nasa et al 2002; Ades et al 2003; Atkins and Walters

2003), for adoptive immunotherapy of malignancy

(Landsteiner and Levine 1929; Landsteiner 1931;

Barrett 2003; Chakrabarti and Childs 2003) and for

reconstitution of ‘normal’ immune function as in

treat-ment of severe combined immunodeficiency (SCID) and

Wiskott–Aldrich syndrome (Filipovich et al 2001).

The role of HLA ‘compatibility’ falls into four different

areas: (1) sufficient compatibility to permit engraftment

and prevent late rejection (with appropriate preparative

and immunosuppressive regimens); (2) enough

com-patibility to minimize graft-versus-host-disease (GvHD);

(3) ample immune reconstitution to permit

immuno-surveillance; and (4) sufficient immune potency to effect

adoptive immune therapy of neoplasia HLA identity is

neither necessary nor sufficient to ensure these effects,

but serological and molecular similarities are the best

available surrogate assays to guide related and unrelated

transplants Both GvHD and graft-versus-tumour (GvL)

occur in the presence of a full HLA match, suggesting

that the classical HLA molecules themselves are not

targets of allosensitization, but rather present

poly-morphic molecules expressed by recipient cells that are

recognized by the grafted immune cells

Graft-versus-host disease: the dark cloud of

haematopoietic cell grafts

Donor lymphocytes engraft, replicate and react against

the normal tissues of the recipient, resulting in a

syn-drome known as GvHD Myeloablative conditioning

administered before transplant effectively minimizes

graft rejection The art of post-transplant

immunosup-pression consists of achieving a balance between graft

immunocompetence and GvHD without allowing

rejection of the graft The risk of GvHD increases with

genetic disparity between donor and recipient HLA

identical twins have the least chance of developing

GvHD, followed by HLA identical siblings, minor

degrees of mismatching among siblings, and unrelated

donors of differing degrees of similarity at the MHC

locus (Longster and Major 1975; Hansen et al 1999).

However, while the genetic homogeneity between donor

and recipient generally decreases the risk of GvHD, it

lessens the therapeutic benefit and increases the chance

of tumour relapse as well (Weiden et al 1981).

Graft-versus-tumor effect: a silver lining of graft-versus-host disease?

One possibly beneficial effect of GvHD, or perhaps

an immunological activity difficult to separate fromGvHD, is rejection of recipient tumour cells by thedonor immune system (graft-versus-leukaemia (GvL)

effect) (Mavroudis and Barrett 1996; Mavroudis et al.

1998) The GvL effect has long been recognized to play

a powerful therapeutic role in the treatment of chronicmyelocytic leukaemia and more recently recognized

as treatment of refractory malignant disorders ing some solid tumours (graft-versus-tumour, GVT)(Childs and Barrett 2002) GVT may be the mostpotent form of tumour immunotherapy currently inclinical use, but its mechanism(s) of action is stillpoorly understood Allogeneic T cells clearly play afundamental role in the initiation and maintenance

includ-of the effect on neoplastic cells (Kolb et al 1990) The

risk of relapse increases markedly for patients withchronic myelogenous leukaemia who received a T-cell-depleted graft compared with a subset of patients whohad received a T-cell-replete one, although the former

patients avoided significant GvHD (Leak et al 1990; Champlin et al 2000) These results suggest that GvHD

is a biological entity different from the GvL effect

In addition, upon leukaemia relapse, administration

of donor lymphocyte infusion can induce clinical and molecular remission Donor T cells may target notonly tumour-specific antigens but also allelic variants

of these antigens, minor histocompatibility antigensand, in the case of HLA-mismatched transplants, HLAantigens disparate from the donor but expressed by thetumour cells (Leddy and Bakemeier 1967; Lederman

et al 1983; Marijt et al 2003) Although several

theories about the mechanisms of the GvL effect havebeen proposed, the reasons that allogeneic T cells seemsuperior to native tumour immunity for some leuk-aemias and solid tumours remain to be clarified

Effect of previous transfusion on success

of bone marrow grafting

Previous transfusions, particularly from close relatives,prejudice the success of subsequent bone marrow

grafting (Storb et al 1980) The chance of rejection of

the graft increases with the number of transfusions

If future recipients of a bone marrow graft need to

be transfused, they should receive leucocyte-depleted

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blood or blood components from random donors

and not from relatives The discrepancy between the

effect of transfusion of blood containing white cells

on grafted bone marrow and the apparent mitigating

effect on renal grafts has not been explained The

development of GvHD after the transfusion of

allo-geneic leucocytes is described in Chapter 15

HLA and organ grafting

Renal grafts

Significance of HLA antibodies When HLA

allo-antibodies directed against antigens expressed on the

donor kidney are present in the serum of a renal graft

recipient, acute or hyperacute rejection of the graft will

occur It is therefore necessary to perform a

cross-match between the patient’s serum and the B and T

lymphocytes of the donor Not all antibodies detected

in the crossmatch are harmful Cold-reacting IgM

autoantibodies directed against B and T cells may be

present in the serum of dialysis patients and do not

appear to be harmful (Ting 1983)

Significance of matching for HLA The extent to

which matching for HLA improves renal graft survival

remains controversial In many studies, matching has

had no obvious benefit due, perhaps, to the small

num-bers of cases studied, interference of the many factors

influencing graft survival and incomplete tissue typing

Furthermore, the survival of mismatched kidneys

has improved greatly following the discovery of the

beneficial effect of previous blood transfusion and of

the value of ciclosporin A as an immunosuppressive

drug Nevertheless, in some large studies, a significant

beneficial effect of HLA matching on long-term graft

survival has been observed

In a study in which 240 laboratories participated,

the results of 30 000 first cadaver kidney transplants

were analysed Cases in which the donor and recipient

were typed for all known ‘splits’ of HLA-A and -B

antigens and those in which typing was restricted to

the broad antigens were analysed separately At 3 years,

an 18% difference in survival rates between grafts with

zero and four mismatches typed for A and B antigen

splits was found, but only a 2% difference when typing

was restricted to broad antigens When A, B and DR

antigens were considered together, the differences in

rates of survival were 31% and 6%, respectively, in the

two groups It was concluded that typing for antigensplits is important (Opelz 1992) Molecular typing of

DR alleles revealed an error rate in serological typing

of about 25% (Mytelineos et al 1990) The impact of

DR matching is particularly significant if patients and

donors are typed at the DNA level (Opelz et al 1993).

In a recent study, complete matching for serologicallydetermined HLA-A, B and DR antigens was found tohave a significant and clinically important impact on

short- and long-term graft survival (Opelz et al 1999).

On the other hand, partial matching provided little

benefit (Held et al 1994) The advantage of complete

matching was diminished by the negative influence oflonger periods of organ preservation and by the factthat in practice only 50% of perfectly matched kidneyswere actually transplanted into the identified recipient

An analysis of more than 150 000 renal transplantsbetween 1987 and 1997 in the Collaborative TransplantStudy indicates that a first cadaver graft with a 6-locusmismatch has a 17% lower 10-year survival than a graft

with no mismatch at these sites (Opelz et al 1999) In

the latter study, the matching effect is even more ing in patients with highly reactive preformed lympho-cytotoxic antibodies Among first cadaver transplantrecipients with antibody reactivity against > 50% ofthe test panel, the difference in graft survival at 5 yearsbetween patients with zero or six mismatches reached30% Once again, correction of serological HLA typ-ing errors by more accurate DNA typing resulted in

strik-a significstrik-antly improved HLA mstrik-atching effect, strik-andmatching for the class II locus HLA-DP, a locus thatcan be typed reliably only by DNA methods, showed

a significant effect for cadaver kidney re-transplants.Non-HLA transplant immunity may be more import-

ant for long-term graft survival (Opelz et al 2005).

For brief discussions of the importance of ABO as amajor histocompatibility system and of the possibleeffects of Lewis groups on renal transplantation, seeChapter 4

Liver grafts and heart–lung grafts

The survival of liver grafts is reportedly improved withHLA matching and is worse when the T-cell cross-

match is positive (Nikaein et al 1994) However, this

finding could not be confirmed in the Collaborative

Transplant analysis (Opelz et al 1999) Matching for

HLA-DR diminished the frequency of rejection episodesafter heart transplantation from 34% to 16%, and at

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3 months there was an additional beneficial effect of

HLA-B matching (Sheldon et al 1994) An independent

study of heart transplants showed a highly significant

impact of HLA compatibility on graft outcome (P<

0.0001) (Opelz et al 1999) In practice, matching for

HLA is much more difficult in the transplantation of

liver and heart than of kidney, mainly because there is

no large pool of HLA-typed recipients to choose from

Immunomodulatory effect(s) of transfusion

As knowledge about the mechanisms of immune

responsiveness and tolerance evolves, and as tools

to measure alterations in immunity become available,

additional immunological consequences of blood

transfusion are being detected Numerous variationsin

circulating blood cells have been reported in patients

transfused with allogeneic blood (see below) Some of

these changes persist for months or even longer after

transfusion The lingering question has been whether

these observations represent no more than laboratory

curiosities, or whether they reflect some clinically

relevant alteration in the recipient’s immune status,the

so-called ‘immunomodulatory effects’ of blood

trans-fusion Based on the sum of clinical evidence (see below)

immunomodulation seems likely to be added to the list

of unintended consequences of allogeneic blood

trans-fusion The magnitude and importance of these effects,

the causative agents, the biological mechanisms and

the patients or patient groups that are at particular risk

have yet to be defined (Klein 1999)

Dzik (2003) has suggested that there may be two

categories of immunosuppressive transfusion effect:

one that is HLA dependent and directed against

adap-tive immunity and a second that is mild, non-specific

and directed against innate immunity The

non-specific effect might result from the infusion of blood

cells that undergo apoptosis during refrigerated

storage The infusion of apoptotic cells has been

shown to be immunosuppressive in animal models

Immunosuppression resulting from the infusion of

apoptotic cells may be linked to transforming growth

factor beta (TGF-β) (Dzik 2003)

Changes in recipient’s lymphocytes after

blood transfusion

Following the transfusion of large amounts of fresh

or stored blood, changes develop in the recipient’s

lymphocytes after an interval of about 1 week Atypicallymphocytes increase by a factor of five or more and lymphocytes may incorporate 3H-thymidine in vitro

at an increased rate Values return to the sion level by about 3 weeks Changes are not seen aftertransfusion of frozen and washed (leucocyte-depleted)

pretransfu-red cells (Schechter et al 1972) Confirmatory

observa-tions were published by Hutchinson and co-workers(1976) The changes are interpreted as a response todonor HLA antigens (presumably of the Dw series)

and may be regarded as those of an MLC in vivo A

number of other alterations of immune cells including

a decrease in NK function and delayed hypersensitivity

have been published (Tartter et al 1986, 1989; Jensen

et al 1992) Blood transfusion alters immune cell

anti-gen expression in premature neonates and may initially

be immunostimulatory and later immunosuppressive

(Wang-Rodriguez et al 2000) Donor lymphocytes

may circulate for prolonged periods in some patientgroups, such as trauma victims, whereas in others such as patients infected with HIV, microchimerism

appears to be transient (Kruskall et al 2001; Lee et al.

1995, 1999, 2001) The immunomodulatory role ofpersistent microchimerism post transfusion and itsrelationship to the HLA system are areas of activeinvestigation For the relationship of microchimerismand transfusion-associated GVHD, see Chapter 15

Effect of previous transfusion on success

of renal graft

Patients who have antibodies against HLA antigens ofthe donor undergo acute rejection of renal allografts

On the other hand, blood transfusion has been shown

to have a striking effect in improving the survival

of subsequent renal grafts in subjects who have notdeveloped cytotoxic antibodies, or who have done soand have received renal grafts from HLA-compatible

donors (Opelz et al 1973; van Hooff et al 1976).

Leucocytes in the donor blood have been found to beessential for the beneficial effect (Persijn 1984).After the introduction of more potent immunosup-pression with ciclosporin, transfusions before cadavergrafting were found to confer little additional benefit

(Kaban et al 1983; Lundgren et al 1986; Opelz 1987),

although a single-centre randomized study attributedpretransplant transfusion benefit to reduced mortality

related to immunosuppression (Vanrenterghem et al.

1994) However, transfusions improved the 1-year

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graft survival rate by 8% (P< 0.01) in recipients of a

one-DR mismatched graft and by 10% (P< 0.01) in

recipients of a two-DR mismatched graft (Iwaki et al.

1990) Two to four transfusions from random donors

were sufficient to obtain this effect This study

con-cluded that despite the use of ciclosporin, the

prac-tice of giving deliberate transfusions before grafting

should not be abandoned In a randomized, controlled

multicentre trial, cadaver graft survival rate was

significantly higher in the 205 recipients who

under-went three pretransplant transfusions than in the 218

patients who did not receive transfusions (Opelz et al.

1997)

When a kidney from a live donor is used, it is

pos-sible to give both transfusion and graft from the same

donor Donor-specific blood transfusions (DSTs) lead

to increased graft survival rates (Salvatierra et al.

1981, 1986; Kaplan 1984) A disadvantage of DST

is that the patient may develop lymphocytotoxic

antibodies against donor HLA antigens In animal

models it was found that heat treatment of the donor

blood (Martinelli et al 1987), or pre-treatment of the

recipient with donor leucocytes coated with

anti-lymphocyte antibody, diminished the chance of such

immunization (Susal et al 1990) Treatment of the

patient with azathioprine also had this effect

(Anderson et al 1982).

Several mechanisms have been suggested to explain

the beneficial effect of previous blood transfusions

on renal graft survival: (1) the induction of increased

suppressor cell activity (Marquet and Heystek 1981;

Quigley et al 1989); (2) decreased natural killer cell

activity (Gascon et al 1984); (3) specific

unresponsive-ness due to idiotype antibodies, which inactivate T-cell

clones (Woodruff and van Rood 1983; Kawamura

et al 1989); (4) impairment of the function of the

mononuclear phagocyte system (MPS) by iron loading

(Akbar et al 1986; de Sousa 1989); (5) deletion of

clones of cells, which are first activated by blood

transfusion and then killed or inactivated by

high-dose immunosuppressive therapy during the anamnestic

response after transplantation (Terasaki 1984); and

(6) the production of non-cytotoxic, Fc

receptor-blocking antibodies (MacLeod et al 1985; Petranyi

et al 1988).

Sharing of MHC antigens between donor and

re-cipient has been found to determine the extent of the

blood transfusion effect The survival of kidney grafts

in recipients who were given transfusions, and who

shared one HLA-DR antigen with the donors, wassignificantly better (81% at 5 years) than in recipientswho were given transfusions from donors mismatchedfor both DR antigens (57% at 5 years), or in recipientswho were not transfused (45%) Immunizationoccurred less frequently in the recipients who shared

one DR antigen with the donor (Lagaaij et al 1989)

In another study, sharing of one HLA haplotype (or

at least one HLA B and DR antigens) between donorand recipients had a mitigating effect because it led to

a specific suppression of the formation of cytotoxic Tlymphocytes (CTLs), i.e to CTL non-responsiveness;recipients of blood from fully identical donors

remained CTL responders (van Twuyver et al 1991).

Furthermore, transfusion of blood from HLA-identicaldonors induces the generation of suppressor cell-independent, high-affinity CTL against donor antigens

(van Twuyver et al 1994) These mitigating and

im-munizing effects are donor specific, but the beneficialeffect of blood transfusion on kidney survival is alsodue to a non-specific effect Transfusion of blood fromdonors who share one HLA haplotype induces a general decrease in the usage of T-cell Vβ families

(Munson et al 1995) These effects are probably due

to the survival of donor lymphocytes in the recipient.Studies in mice have shown that sharing of H2 anti-gens between donor and recipient of a blood trans-fusion facilitates the persistence of donor lymphoid cells in the recipient, which is associated with tolerancefor donor alloantigens Donor lymphocytes can bedetected 10 –20 years after transplantation in patients

in whom the graft survives for long periods of time.Such chimerism may be important in modulating the

immune response (Starzl et al 1992).

Effect of transfusion on tumour growth and recurrence of cancer

A retrospective analysis of the recurrence rate of cinoma of the colon after surgical resection first suggested that the 5-year disease-free survival rate was reduced by blood transfusion given at the time ofsurgery (Burrows and Tartter 1982) However, despitenumerous subsequent reports, including more than

car-100 observational studies and three controlled trials,and several meta-analyses, the relationship betweenblood transfusion, cancer growth and cancer-free sur-vival remains murky and contradictory (Klein 2001).The numerous variables including different tumours,

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locations, extent of disease, histological grade and

modes of treatment make this a particularly difficult

area to evaluate

Prospective randomized clinical studies have been

conducted with patients undergoing surgery for

colo-rectal carcinoma In one large multicentre

random-ized trial, patients who were operated on because of

colorectal cancer and who needed blood transfusion

were randomized to an autologous or allogeneic

trans-fusion regimen At study conclusion, patients received

allogeneic blood only (133), autologous and

allo-geneic blood (66), autologous blood only (112) or no

blood at all (164) There were no significant

differ-ences between the groups receiving allogeneic blood

or autologous blood only: at 4 years, survival rates

were 67% and 62%, respectively, and in survivors, no

recurrence of cancer in 63% and 66% respectively

On the other hand, many patients did not receive the

‘treatment’ specified by their prospective treatment

assignment, and cancer recurred significantly more

fre-quently in the transfused than in the non-transfused

patients This difference may have been associated

with the circumstances that necessitated transfusion

(Klein 1999) The red cells transfused in the allogeneic

arm were buffy coat depleted as was the routine in the

Netherlands at the time In a second controlled study

of colorectal cancer in the Netherlands, leucoreduced

red cells were compared with buffy coat-reduced red

cells No significant differences were found between

the two trial transfusions in survival, disease-free

sur-vival or cancer recurrence rate after an average

follow-up of 36 months Patients who had a curative resection

and who received blood of any sort had a lower 3-year

survival than non-transfused patients (69% vs 81%,

P= 0.001) These observations confirm an association

between blood transfusion and poor patient survival,

but suggest that the relation is not due to promotion of

cancer (Houbiers et al 1994) The third prospective

study of colorectal cancer from a single centre in

Germany found that blood transfusion was an

inde-pendent factor associated with tumour recurrence,

and that survival of transfused patients tended to be

shorter although the difference was not statistically

significant (Heiss et al 1994) There may well be some

subset of patients, perhaps defined by immune status

or tumour subtype, that is particularly susceptible to

the effects of allogeneic transfusion Demonstration of

such a difference will probably require a large,

care-fully controlled, prospective study

Effect of transfusion on postoperative infections

As is the case with cancer, a large number of tional studies find an association between allogeneicblood transfusion and postoperative bacterial infection,while a few do not (Vamvakas and Blachman 2001) Forexample, Carson and co-workers (1999) conducted aretrospective cohort study of 9598 consecutive patientswith hip fracture who underwentsurgical repair between

observa-1983 and 1993 at 20 hospitals across the USA Bacterialinfection, defined as bacteraemia, pneumonia, deepwound infection or septic arthritis/osteomyelitis wasthe primary endpoint and numerous variables wereincluded in the statistical model; a highly significantassociation was found between serious postoperativeinfection and transfusion Chang and co-workers (2000)analysed a database of 1349 patients undergoing elective colorectal surgery for any disease of the colon

or rectum at 11 university hospitals across Canada Toadjust for confounding effects associated with remoteinfections such as pneumonia and urinary tract infec-tions, the study limited the analysis to postoperativewound infection Allogeneic blood transfusion was ahighly significant independent predictor of postoperat-ive wound infection Vamvakas and Carven (1998)reported a retrospective cohort study of 416 coronaryartery bypass graft patients admitted to one hospital.The endpoints were limited to postoperative woundinfection or pneumonia, and adjustment was made forthe effects of chronic systemic illness and specific riskfactors for wound infection or pneumonia The risk ofpostoperative wound infection or pneumonia increased

by 6% per unit of allogeneic red blood cells (RBCs) and/

or platelets transfused, or by 43% for a patient receivingthe mean transfusion dose of 7.2 units of either compon-ent Nevertheless, these analyses are inevitably flawed,despite meticulous multivariate testing, by the numerousvariables that predispose to postoperative infection(comorbidities, catheters, respirator time, impairedconsciousness, etc.), not to mention factors related tothe blood components such as storage time and method

of preparation (Vamvakas and Carven 1998)

Seven randomized controlled trials compare theincidence of postoperative infection between recipi-

ents of buffy coat-reduced (Heiss et al 1993; Busch

et al 1994; Houbiers et al 1994; Jensen et al 1996; van de Watering et al 1998) or standard allogeneic red cells (Tartter et al 1998) or whole blood (Jensen et al.

1992) and recipients of autologous or WBC-reduced,

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buffy coat-reduced allogeneic red cells or whole blood.

Two studies (Jensen et al 1992, 1996) reported a

significant effect, two studies (Heiss et al 1993; van

de Watering et al 1998) reported a marginal effect,

and three studies (Busch et al 1994; Houbiers et al.

1994; Tartter et al 1998) did not detect an effect

The strengths and weakness of these studies have

been analysed exhaustively (Vamvakas and Blachman

2001) However, insufficient data are available to

perform the kind of meta-analysis that might help

draw conclusions from these studies

Postoperative mortality

In addition to the possible association between

allo-geneic transfusion and postoperative infection, van de

Watering and co-workers (1998) detected an

unex-pected association between WBC-containing allogeneic

blood transfusion and postoperative mortality from

causes other than postoperative infection In total,

24 out of 306 patients (7.8%) transfused with buffy

coat-reduced red cells died, compared with 11 out of

305 patients (3.6%) receiving buffy coat-reduced red

cells that were leucoreduced before storage, and 10 out

of 303 patients (3.3%) receiving buffy coat-reduced

red cells that were leucoreduced after storage (P= 015)

The overall difference in 60-day mortality was due to a

highly significant difference among the three

random-ization arms The number of RBC units transfused

was the most significant predictor of postoperative

mor-tality The association between leucocyte-containing

allogeneic blood and increased mortality may be

lim-ited to cardiac surgery and should not be extended to

other clinical settings At the very least, the finding

requires confirmation by a study designed with

mor-tality as the primary endpoint

Possible role of HLA in habitual abortion

Parental sharing of HLA antigens has been thought to

be a cause of habitual abortion In such cases,

non-cytotoxic antibodies that are normally produced in the

mother and that protect the fetus are absent (Adinolfi

1986; Scott et al 1987).

Immunization of women with leucocytes has been

employed with the object of correcting the

immuno-logical unresponsiveness (Taylor and Falk 1981; Beer

et al 1985) One problem in assessing the benefit of

such immunization is that the definition of habitual

abortion varies Furthermore the chance of a successfulpregnancy after three abortions is about 60% (Regan1991) One prospective randomized trial (Mowbray

et al 1987) has shown an apparent benefit; in another

trial, no clear advantage of leucocyte injection wasobserved, and the authors expressed their concernabout severe growth retardation seen in some fetuses

(Beer et al 1985) The Recurrent Miscarriage Study

enrolled women who had had three or more eous abortions of unknown cause in a double-blind,

spontan-multicentre, randomized clinical trial (Ober et al.

1999) In total, 91 women were assigned to tion with paternal mononuclear cells (treatment) and

immuniza-92 to immunization with sterile saline (control) Theprimary endpoints were the inability to achieve preg-nancy within 12 months of randomization, or a preg-nancy that terminated before 28 weeks of gestation(failure); and pregnancy of 28 or more weeks of gesta-tion (success) Immunization with paternal mono-nuclear cells did not improve pregnancy outcome

in women with unexplained recurrent miscarriage.However, it is possible that a subset of respondersmight be identified by using some as yet unrecognizedlaboratory determination or susceptibility factor.Until this is possible, immune therapy in women withhabitual abortion should be restricted to clinical trials

(Moloney et al 1989).

Tests for HLA alleles, antigens and antibodies

HLA allelesHLA alleles can be determined directly at the DNAlevel The resolution of DNA-based typing is limitedonly by the available allele-specific probes The relev-ant techniques are based on several different prin-ciples (see Chapter 3) While the heterogeneity of theMHC has made high-resolution typing problematicfor matching donor and recipient for transplantation

(Petersdorf et al 2001), the stringency of the HLA role

in antigen presentation has made high resolutionincreasingly desirable for immunotherapy trials.Anthony Nolan HLA informatics group publishes up-to-date online HLA Class I and II Sequence Alignments(http://www.anthonynolan.com/HIG/data.html)

Sequence-specific oligonucleotides DNA is amplified

in the polymerase chain reaction (PCR) and a set of

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sequence-specific oligonucleotides (SSOs) is used in a

dot-blot or reverse dot-blot hybridization technique

to detect allelic sequences (Saiki et al 1986, 1989; Ng

et al 1993) Several modifications of this technique

have been described (Bidwell 1994) In one, a strand of

heat-denatured, amplified DNA is ligated with SSOs

by an added ligase Ligation only occurs when the

sequences of the DNA and SSO are identical The ligated

product is detected by enzyme-linked immunosorbent

assay (ELISA) (Fischer et al 1995) The techniques

permit the identification of alleles, even of those

differ-ing from each other by a sdiffer-ingle nucleotide

Sequence-specific primers PCR is performed with a

set of sequence-specific primers (SSPs) that will only

amplify DNA with sequences complementary to the

primers (Olerup et al 1993; Olerup 1994; Bunce et al.

1995) A simple and quick SSP test in microplates has

been described (Chia et al 1994).

The limitations of both SSO and SSP are

require-ments for a large number of PCRs to include the known

alleles, and the inability to identify polymorphisms

unless the variation happens to lie within the region

spanned by the assay These limitations are addressed

by nucleotide sequencing of PCR-amplified DNA, the

method of choice for ‘high-resolution’ typing that is

required in the selection of an unrelated stem cell donor

(Spurkland et al 1993) High-throughput robotic

sequence-based typing allows daily sequencing of

hundreds of genomic fragments, and high-density

array technology promises to permit extensive typing

of polymorphisms, both known and unknown, on

microchips (Adams et al 2001; Wang et al 2003).

HLA antigens and antibodies

Class I antigens are determined using the

lymphocyto-toxicity test Crossreactivity and the lack of specific

antisera led to difficulties in HLA typing; several antisera

must be used in typing for a particular antigen In

sero-logical typing for DR and DQ antigens, the two-colour

fluorescence test or the lymphocytotoxicity test on B

cells is applied (see below) These same techniques are

used for the detection of class I and class II antibodies

respectively Lymphocytotoxicity is declining in interest

in the USA as most laboratories switch to easier, higher

resolution molecular methods However,

immunolo-gical methods remain valuable to characterize

func-tional aspects of HLA as molecular methods cannot

define whether an HLA allele is expressed or how asequence correlates with empirically determined anti-gen importance

Lymphocytotoxicity test

Complement-dependent cytotoxicity remains the standard test for determining HLA class I antigens.Lymphocytes are incubated with antibody and rabbitcomplement, and a dye (Trypan blue or eosin) is thenadded If the lymphocytes carry an antigen corres-ponding to the antibody, complement is fixed, the cell membrane is damaged and dye enters and stainsthe cell (blue or red) The percentage of stained cells iscounted Live cells are unstained, smaller and refrac-tile It is essential to use a pure lymphocyte suspension,

as platelets carry A, B and C antigens and granulocytesare always killed in the cytotoxic assay and stain non-specifically Details of the NIH-recommendedlymphocytotoxicity test, using microdroplets, werepublished by Terasaki and co-workers (1973)

For the determination of DR and DQ antigens bylymphocytotoxicity, B lymphocytes can be isolated:(1) by removing the T lymphocytes from a lymphocytesuspension by rosetting with Z-aminoethylisothiouro-nium bromide-treated sheep red cells and centrifuga-tion on Ficoll-hypaque (density 1.077) (Pellegrino

et al 1976); (2) by the use of nylon fibre columns (Wernet et al 1977); or (3) by the use of magnetic

beads coated with monoclonal antibodies specific for

class II epitopes (Vartdal et al 1986).

In the two-colour fluorescence tests the IgG on the Bcells is capped with FITC-labelled anti-IgG followed

by a cytotoxicity test The B cells can be distinguishedfrom the T cells by the green IgG cap on their surface.For a description of the technique, see van Rood andvan Leeuwen (1976)

The mixed lymphocyte culture

This test was described by Bain and co-workers(1964) The principle is to irradiate or add a substancesuch as mitomycin C to one sample, usually thedonor’s, and to mix these lymphocytes with thosefrom another subject such as a potential recipient.Irradiation, or treatment with mitomycin C, preventslymphocytes from transforming to blast cells but doesnot destroy their ability to stimulate other lymphocytes.Blast transformation of the untreated lymphocytes

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indicates that they have recognized a foreign antigen

on the treated lymphocytes (Bach and Voynow 1966),

and this transformation can be assessed by measuring

the incorporation of tritiated thymidine (one-way

MLC)

In the MLC, the cells that stimulate are B cells

and monocytes carrying Dw determinants and class II

antigens Those that respond are T cells (Potter and

Moore 1977)

If irradiated or mitomycin C-treated stimulator

cells, homozygous for a Dw determinant (homozygous

typing cells, or HT), are used they can only stimulate

untreated lymphocytes that do not carry the Dw

deter-minant for which they are homozygous Thus, panels

of HTC are used to identify Dw determinants (Bradley

et al 1972).

The two-way MLC, in which the lymphocytes in

both samples are able to respond by blast formation,

has been used as a final test for HLA identity of donors

and recipients of bone marrow who are serologically

identical

HLA antibody detection

Typed repository cell lines are used in a

complement-dependent cytotoxicity assay to identify alloantibodies

in sera of sensitized subjects The percentage of cell

lines killed by the sera is used as a rough measure of

the degree of sensitization or ‘panel reactive antibody’

(PRA) reactivity Some antibodies activate complement,

yet kill cells inefficiently, a phenomenon known as

‘cytotoxicity negative absorption positive’ (CYNAP)

(Lublin and Grumet 1982) The CYNAP phenomenon

may result in underestimation of sensitization

How-ever, augmentation of the assay to increase sensitivity

may implicate innocuous antibodies and thus

over-estimate clinically relevant sensitization Another

method of identifying alloantibodies uses (Le Pendu

et al 1986; Le Pont et al 1995) flow cytometry of

a variety of microbeads loaded with known HLA

alleles (Guertler et al 1984; Moses et al 2000) An

interlaboratory comparison of techniques suggests

that considerable inconsistencies in serum screening

and crossmatching exist among laboratories

particip-ating in the American Society for Histocompatibility

and Immunogenetics/College of American Pathologists

surveys (Duquesnoy and Marrari 2003) The lack of

uniformity in test results may limit the usefulness of

these methods in a clinical setting

Other antigens found on leucocytes

Some red cell antigens are also found on leucocytes; see Chapters 4 and 6

Antigens located on granulocytes (humanneutrophil antigens)

Nomenclature: confusion, controversial and evolving

Neutrophil antigens were first characterized using seracollected from neutropenic patients who formed clin-ically important allo- and autoantibodies Althoughthe presence of the first granulocyte-specific antigen,NA1, was inferred from the presence of an antibody in

a case of neonatal neutropenia in 1960, the antigen

was not identified until 1966 (Lalezari et al 1960;

Lalezari and Bernard 1966a) As new antigens werediscovered, nomenclature threatened to assume some

of the quirky randomness that characterized red cellblood group antigens A new nomenclature was pro-posed in 1998 by an International Society of BloodTransfusion (ISBT) Working Party to permit separatenotations for the phenotype associated with the glyco-protein location and for the alleles, according to theguidelines for human gene nomenclature (Bux 1999).Although the proposed nomenclature has been criti-cized for including antigens found on cells other thangranulocytes, the ISBT proposal represents a soundfirst attempt at standardization

In the ISBT nomenclature, antigen systems arereferred to as human neutrophil antigens (HNA) The antigen systems, the polymorphic forms of theimmunogenic proteins, are indicated by integers andspecific antigens within each system are designatedalphabetically by date of publication Alleles of thecoding genes are named according to the Guidelinesfor Human Gene Nomenclature Neutrophil antigensNA1 and NA2 became HNA-1a and HNA-1b in thenew nomenclature and the third antigen reported,NB1 became HNA-2a (Table 13.1)

The HNA-1 system

The neutrophil-specific antigens HNA-1a and -1b(NAl and NA2) are products of alleles that form a biallelic system confined to granulocytes (Lalezari

et al 1960; Lalezari and Radel 1974) and NK cells.

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Exceptions to the inheritance of the NA antigens first

suggested the possibility of a silent allele at the NA

locus (Lalezari et al 1975; Clay 1985) The HNA-1

antigens are located on the FcγRIIIb of neutrophils

(Huizinga et al 1990) FcγRIIIb on neutrophil

mem-branes is a phosphatidylinositol-linked glycoprotein

with a molecular weight of 50 –80 kDa (Huizinga et al.

1989, 1990)

FcγRIIIb and the HNA-1 antigens are encoded by

the FCGR3B gene located on chromosome 1q23–24

within a cluster of two families of the FcγR genes,

Fc γR2 and FcγR3 The FcγR3 family is made up of

FCGR3A and FCGR3B FCGR3B is highly

homo-logous to FCGR3A, which encodes FcRIIIa The most

important difference between the two genes is a C-to-T

change at 733 in FCGR3B, which creates a stop codon

in FcγRIIIb As a result, FCGR3A has 21 more amino

acids than FCGR3B, and FCGR3A is a

transmem-brane rather than a GPI-anchored glycoprotein (Ory

et al 1989; Ravetch and Perussia 1989; Huizinga et al.

1990; Trounstine et al 1990) FcγRIIIa is expressed

only by NK cells and FcRIIIb only by neutrophils

(Trounstine et al 1990).

HNA-1a, -1b and –1c polymorphisms The HNA-1

antigen system consists of the three alleles HNA-1a,

-1b and -1c (Bux et al 1997) The antigens are also

known as NA1, NA2 and SH (Table 13.1) The gene

frequencies of the three alleles vary widely among

dif-ferent racial groups (Hessner et al 1999; Matsuo et al.

2001) Among white people, the frequency of the gene

encoding HBA-1a, FCGR3B*1, is between 0.30 and

0.37, and the frequency of the gene encoding HNA-1b,FCGR3B*2, is from 0.63 to 0.70 In Japanese andChinese populations, the FCGR3B*1 gene frequency

is from 0.60 to 0.66, and the FCGR3B*2 gene quency is from 0.30 to 0.33 The gene frequency of the gene encoding HNA-1c, FCGR3B*3, also variesamong racial groups FCGR3B*3 is expressed by neutrophils from 4% to 5% of white people and 25

fre-to 38% of African Americans (Kissel et al 2000).

The FCGR3B*1 gene differs from the FCGR3B*2gene by only five nucleotides in the coding region at

positions 141, 147, 227, 277 and 349 (Ory et al 1989; Ravetch and Perussia 1989; Huizinga et al 1990; Trounstine et al 1990) Four of the nucleotide changes

result in changes in amino acid sequence between theHNA-1a and the HNA-1b forms of the glycoprotein.The fifth polymorphism at site 147 is silent The gly-cosylation pattern differs between the two antigensbecause of the two nucleotide changes at bases 227

and 277 The HNA-1a form of Fc γRIIIb has six

N-linked glycosylation sites and the HNA-1a form hasfour glycosylation sites

The gene encoding the HNA-1c form of F γcRIIIb,

FCGR3B*3, is identical to FCGR3B*2 except for

a C-to-A substitution at amino acid 78 of FcRIIIb (Bux et al 1997) In many cases, FCGR3B*3 exists

on the same chromosome with a second or duplicate

FCGR3B gene (Koene et al 1998).

Several other sequence variations in FCGR3B havebeen described These chimeric alleles have single-basesubstitutions involving one of the five SNPs that

distinguish FCGRB3B*1 and FCGR3B*2 FCGR3B

Antigen system Antigens Location Former name Alleles

CR3, C3bi receptor; gp, glycoprotein; HNA, human neutrophil antigen; ISBT,

International Society of Blood Transfusion; LFA-1, leukocyte function antigen-1.

From Wang E, Marincola FM, Stroncek D Human leukocyte antigen (HLA) and

human neutrophil antigen (HNA) systems In: Hematology: Basic Principles and

Practice (2005), Philadelphia, PA: Elsevier Churchill Livingstone.

Table 13.1 ISBT Human Neutrophil

Antigen (HNA) nomenclature.

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alleles that more closely resembled FCGR3B*2 were

found more often in African Americans than in white

people or Japanese people (Matsuo et al 2001).

Function of HNA-1 antigens The low-affinity

FcγRIIIb receptors link humoral and cellular immune

function The FcγRIIIb on effector cells bind cytotoxic

IgG molecules and immune complexes containing

IgG Polymorphisms in FcγRIIIb affect neutrophil

func-tion Neutrophils that are homozygous for HNA-1a

have a greater affinity for IgG3 than do those that

are homozygous for HNA-1b (Nagarajan et al 1995).

Neutrophils from subjects homozygous for HNA-1b

phagocytize erythrocytes sensitized with IgG1 and

IgG3 anti-Rh monoclonal antibodies and bacteria

opsonized with IgG1 less efficiently than do

granulo-cytes homozygous for HNA-1a (Bredius et al 1994).

Fc γRIIIb deficiency Blood cells from patients with

paroxysmal nocturnal haemoglobinuria (PNH) lack

the GPI-linked glycoproteins and their granulocytes

express reduced amounts of FcγRIIIb and the HNA-1

antigens (Huizinga et al 1990) Genetic deficiency of

granulocyte FcγRIIIb and the HNA-1 antigens has

been reported With inherited deficiency of FcγRIIIb,

the FCGR3B gene is deleted along with an adjacent

gene, FCGR2C (De Haas et al 1995) Despite the

prim-ary role of FcγRIIIb in neutrophil function, deletion

of the entire Fc γRIIIB gene results in no obvious

clin-ical abnormality Although most subjects who lack

FcγRIIIb appear healthy, too few have been studied

to ensure that no subtle alteration in immune

func-tion is present In a study of 21 FcγRIIIb subjects

with FcγRIIIb deficiency, two were found to have

autoimmune thyroiditis and four had sustained

mul-tiple episodes of bacterial infections (De Haas et al.

1995)

FCGR3B polymorphisms and disease

associations

Several studies suggest that FCGR3B polymorphisms

affect the incidence and outcome of some autoimmune

and inflammatory diseases Children with chronic

immune thrombocytopenic purpura were more likely

to be FCGR3B*1 homozygous than were control

sub-jects (Foster et al 2001), but Spanish patients with

systemic lupus erythematosus were more likely to be

FCGR3B*2 homozygous (Gonzalez-Escribano et al.

2002) Myasthenia gravis is more severe in FCGR3B*1

homozygous patients (Raknes et al 1998), but multiple

sclerosis is more benign in FCGR3B*1 homozygous

patients (Myhr et al 1999) Patients with chronic

granulomatous disease who are FCGR3B*1 zygous are less likely to develop major gastrointestinal

homo-or urinary tract infectious complications comparedwith those who are heterozygous or FCGR3B*2 homo-

zygous (Foster et al 1998) As FCGR3B is clustered

with FCGR3BA and FCGR2B on chromosome 1q22,some of these findings may reflect in part linkage dis-equilibrium among Fc receptors

The HNA-2 system

The HNA-2 system has one well-described allele,HNA-2a (NB1) expressed only on neutrophils, meta-

myelocytes and myelocytes (Stroncek et al 1998a).

The 58- to 64-kDa glycoprotein that carries HNA-2a(NB1 gp) is located on neutrophil plasma membranesand in secondary granules, and is linked to the plasmamembrane by a glycosylphosphatidylinositol (GPI)anchor HNA-2a is expressed on 45–65% of circulat-ing neutrophils; expression is greater on neutrophils

from women than from men (Stroncek et al 1996; Matsuo et al 2001) Pregnant women express HNA-

2a more strongly than do healthy female blood

donors (Caruccio et al 2003) Expression of HNA-2a

decreases with age in women, but remains constant

in men Administration of G-CSF to healthy subjects can increase the proportion of neutrophils express-

ing HNA-2a to near 90% (Stroncek et al 1998b).

Monoclonal antibodies specific to HNA-2a have beenclustered as CD177 The role of CD177 in neutrophilfunction is unknown Women who lack NB1 gp arehealthy Although the expression of HNA-2a is reduced

on neutrophils from patients with PNH and chronicmyelocytic leukaemia (CML), no clinical significancehas been attributed to this observation

HNA-2 polymorphisms HNA-2a is expressed on

neutrophils by approximately 97% of white people,95% of African Americans and 89–99% of Japanese

people (Matsuo et al 2000; Taniguchi et al 2002).

HNA-2a has been reported to have an allele, NB2, but the product of this gene cannot be identified reli-ably with alloantisera, and no monoclonal antibody

specificity for NB2 has been identified (Stroncek et al.

1993a) The HNS-2a-negative neutrophil phenotype is

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due to a CD177 transcription defect Kissel HNA-2a

genes from two women with HNA-2a-negative

neu-trophils, who produced HNA-2a-specific

alloanti-bodies have been studied and CD177 cDNA sequences

were present in both women Sequencing of cDNA

prepared from neutrophil mRNA demonstrated

acces-sory sequences in the lengths coding CD177

The HNA-3 system

The HNA-3 antigen system has one antigen HNA-3a

that was previously known as 5b HNA-3a is

expressed by neutrophils, lymphocytes, platelets,

endothelial cells, kidney, spleen and placental cells

(van Rood and Ernisse 1968) and weakly expressed on

red cells (Rosenfield et al 1967) The gene encoding

HNA-3a is located on chromosome 4 (van Kessel et al.

1983), but has not yet been cloned The nature and

function of the 70 –95 kDa gp is unknown Potent

anti-HNA-3a agglutinins in transfused plasma can cause

transfusion-related acute lung injury (TRALI) (see

Chapter 15)

HNA-4 and HNA-5 systems

The HNA-4 and HNA-5 antigens are located on the β2

integrins Each system contains only a single antigen,

HNA-4a and HNA-5a The HNA-4a antigen,

previ-ously known as Marta, has a phenotype frequency of

99.1% in white people (Kline et al 1982) HNA-4a is

expressed on granulocytes, monocytes and

lympho-cytes, but not on platelets or red blood cells HNA-4a

has been located on the αM chain (CD11b) of the

receptor CR3 Neonatal alloimmune neutropenia has

been caused by antibody to HNA-4a, but this is the

exception rather than the rule (Fung et al 2003) A

second polymorphism of the β2 integrins, HNA-5a,

previously Ondawith a frequency of 95%, is located

on the α-chain of the leucocyte function-associated

antigen (LFA-1, CD11a) molecule (Simsek et al.

1996)

Other granulocyte-specific antigens

Other granulocyte-specific antigens are ND1, NE1

and LAN (Lalezari and Radel 1974; Verheugt et al.

1978; Claas et al 1979; Rodwell et al 1991) Like the

NA antigens, LAN is located on the FcγRIIIb (Metcalfe

and Waters 1992) Another high-frequency antigen,

also located on the FcγRIIIb, was described by Bux and colleagues (1994) The antigen NC1 (Lalezari

and Radel 1974) is identical with HNA-1b (Bux et al.

1995a) Most granulocyte-specific antigens have beendefined by alloantibodies, but ND1 and NE1 weredefined by autoantibodies These granulocyte-specificantigens appear to be true differentiation antigens, asthey appear at the myelocyte or metamyelocyte stage

or even later (Lalezari 1977; Evans and Mage 1978)

Antigens on granulocytes and monocytes

The following antigens have been shown to be present

on granulocytes and monocytes: HGA-1 (Thompson

et al 1980) and the HMA-1 and HMA-2 antigens, products of a biallelic gene (Jager et al 1986) The

AYD antigen is shared by granulocytes, monocytesand endothelial cells (Thompson and Severson 1980).The 9a antigen, which was first thought to be granulo-cyte specific, is also expressed on monocytes (Jager

et al 1986).

Antibodies to neutrophil antigens

Antibodies to neutrophil antigens may be responsiblefor five different clinical syndromes: (1) neonatal allo-and isoimmune neutropenia; (2) autoimmune neutro-penia; (3) transfusion-related alloimmune neutropenia;(4) pulmonary infiltrates following transfusion (TRALI);and (5) febrile reactions following transfusion Thelast two conditions are discussed in detail in Chapter 15.Transfusion-related alloimmune neutropenia is prob-ably a variant of TRALI and appears to be rare (Wallis

et al 2002).

Neonatal alloimmune neutropenia

This syndrome, analogous to haemolytic disease of thenewborn (HDN), is usually recognized when infection

in a newborn infant is found to be accompanied bysevere neutropenia Neutrophil antigens in the fetusthat are inherited from the father but foreign to themother provoke formation of maternal IgG antibodiesthat cross the placenta and react with the neonate’sneutrophils (Lalezari and Bernard 1966b) Absoluteneutrophil counts typically range from 0.100 to 0.200

× 109/l Bone marrow examination reveals myeloidhyperplasia The syndrome is self-limited, but maypersist from days to weeks as passive antibody is

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cleared (Bux et al 1992) Treatment with IVIG or

recombinant cytokines such as granulocyte

colony-stimulating factor (G-CSF) has met with variable

success (Maheshwari et al 2002).

In reviewing the syndrome, Lalezari and Radel

(1974) described results in 19 infants from 10 families

The specificity of the antibody in three families was

HNA-1a; in two, HNA-1b; in four,

anti-HNA-2a; and in one, not determined In four of the

families the first-born infant was affected Antibodies

of other specificities have been implicated, but much

less frequently (Stroncek 2002)

A prospective survey of some 200 pregnant women,

either primiparae at term or multiparae, in which

the woman’s serum was tested against her partner’s

granulocytes and lymphocytes, indicated that the

incidence of neutrophil antibodies was about 3%

(Verheugt et al 1979) The incidence of diagnosed

cases of neonatal alloimmune neutropenia is much

lower

Neonatal isoimmune neutropenia

Subjects who lack a membrane glycoprotein due to

deletion of the gene encoding the glycoprotein may

form strong antibodies (named isoantibodies) against

non-polymorphic determinants on the glycoprotein

Severe neonatal neutropenia caused by maternal

isoantibodies may occur in infants from mothers with

a deletion of the FcγRIIIb gene (Huizinga et al 1990;

Stroncek et al 1991; Cartron et al 1992; Fromont

et al 1992).

Autoantibodies to granulocytes

The first convincing case that implicated

autoanti-bodies as the cause of neutropenia involved a female

infant who had severe infections and was found at the

age of 7 months to have a neutrophil count of 1.0 × 109/l

The peripheral blood contained fewer than 3% mature

neutrophils and the bone marrow revealed virtually no

mature granulocytes, although it did contain normal

granulocyte precursors The patient’s serum contained

the neutrophil-specific autoantibody anti-HNA-1b

with a titre of 16–256 The antibody was mainly IgG

and the patient was HNA-1b positive After steroid

therapy, the granulocyte count rose to 310 × 109/l and

the leucoagglutinin titre fell to 2, but the patient

relapsed when steroids were discontinued (Lalezari

et al 1975) The syndrome is now well established (McCullough 1988; Bux et al 1998).

Autoimmune neutropenia (AIN) in children is tionally divided into two forms In so-called ‘primary’AIN, neutropenia is the sole abnormality and,although neutrophil counts may fall below 0.500/l,bacterial infections, when they occur, are generallybenign Primary AIN is commonly diagnosed betweenthe ages of 5 and 15 months, but has been observed as

tradi-early as day 33 of life (Bux et al 1998) Spontaneous

remission occurs in 95% of the patients within 7–24months A high percentage of autoantibodies (35–86%)bind preferentially to granulocytes from HNA-1a andHNA-1b homozygous donors, but other specificities

have been found (Bux et al 1998; Bruin et al 1999).

The bone marrow is typically normocellular or hypercellular, with a variably diminished number ofsegmented granulocytes For severe infections or prior to surgery, G-CSF, corticosteroids and IVIG (19 mg/kg per day) can effect neutrophil increases of50–100% and each has been used successfully in many

cases (Pollack et al 1982; Bussel and Lalezari 1983; Bux et al 1998).

Secondary AIN occurs in association with otherautoimmune diseases In contrast with primary AIN,infections are usually more severe and the autoanti-body is more commonly directed against FcγRIIIb

(Shastri and Logue 1993; Bruin et al 1999).

Drug-induced immune granulocytopenia

Mechanisms responsible for drug-induced immuneneutropenia are similar to those involving red cells (see Chapter 7) The classic case of pyramidon-induced granulocytopenia described by Moeschlin and Wagner (1952) is an example of the mechanism

in which the drug does not bind firmly to the cells, but in which drug, antibody and a determinant on the cell membrane form a trimolecular complex.Although quinine is usually implicated in drug-induced thrombocytopenia, it may be involved rarely

in cases of drug-induced neutropenia in which the quinine antibodies react with the same glycoprotein

as anti-HNA-2a, and/or an 85-kDa glycoprotein

(Stroncek et al 1994) Drug-induced neutrophil

anti-bodies may be directed against a metabolite of the

drug (Salama et al 1989) Recombinant G-CSF has

reportedly shortened the period of granulocytopenia

in some of these cases

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Reactions to granulocyte transfusions

Granulocyte transfusion recipients sometimes produce

antibodies specific to HNA-1a, HNA-1b and HNA-2a

(see also Chapters 14 and 15) Further transfusion of

granulocytes to patients with these antibodies may

lead to severe febrile and pulmonary transfusion

reac-tions (Stroncek 1996) Haematopoietic progenitor cell

transplant recipients who produce HNA-2a antibodies

as a result of granulocyte transfusions have

experi-enced marrow graft failure (Stroncek et al 1993b).

Tests for granulocyte antibodies and antigens

The following techniques are used in detecting

granulocyte-specific antibodies: (1) granulocyte

agglutination; (2) immunofluorescence; (3)

chemilu-minescence; and (4) monoclonal antibody-specific

immobilization of granulocyte antigen assays

The granulocyte agglutination technique

Pure granulocyte suspensions are prepared by dextran

sedimentation followed by centrifugation of the

super-natant on Ficoll-hypaque (density 1.077) The

con-taminating red cells in the granulocyte pellet at the

bottom of the tube are lysed with ammonium chloride

or distilled water Alternatively, granulocytes can

be isolated by double-density gradient

centrifuga-tion Agglutination techniques are carried out in

microplates

Granulocytes, in contrast to red cells and platelets,

are agglutinated by two different mechanisms:

1 Like red cells and platelets granulocytes are

agglu-tinated by crosslinking of cells by IgM antibodies

2 An entirely different mechanism is responsible for

agglutination of granulocytes by IgG antibodies In

this case, agglutination results from a response to

sensitization by an antibody that requires active cell

participation Sensitization does not lead to immediate

agglutination but to the formation of pseudopods

The granulocytes migrate towards each other until

membrane contact is established (Lalezari and Radel

1974) This process is time and temperature (37°C)

dependent Agglutination may be due to changes in

membrane-bound molecules that cause granulocytes

to adhere to each other, or to IgG antibodies on one

granulocyte that adhere to Fc receptors on other

gran-ulocytes In any case, both IgM and IgG antibodies can

be detected by the granulocyte agglutination test Bothgranulocyte-specific and HLA-A, -B and -C antibodiesare detected, but HLA antibodies are better detected

by the lymphocytotoxicity test

Granulocyte immunofluorescence technique

Purified suspensions of granulocytes are prepared

as described above The granulocytes are fixed withparaformaldehyde, incubated with the serum to

be tested, then washed and finally incubated withfluorescein isothiocyanate-labelled anti-Ig serum TheFab or F(ab′)2fragments of the IgG fraction of anti-human Ig are used because whole IgG anti-Ig tends

to bind to the Fc receptor on granulocytes With theabove modifications, the fluorescein-labelled anti-globulin test is more sensitive than granulocyte agglu-tination for the detection of IgG antibodies (Verheugt

et al 1977).

Using flow cytometry for the granulocyteimmunofluorescence technique (GIFT) instead ofmicroscopy, there is no need for isolating granulo-cytes, as granulocytes can be identified according

to light scatter patterns Furthermore, granulocytes,platelets and lymphocytes can be tested simultane-

ously (Robinson et al 1987) Flow cytometry has been

found to be slightly more sensitive for the detection

of granulocyte antibodies than the GIFT (Sintnicolaas

et al 1991).

Both granulocyte-specific and HLA-A, -B and -Cantibodies are detected in the GIFT The lymphocyto-toxicity test (LCT) and the immunofluorescence testare more sensitive for the detection of HLA class I antibodies If a serum is negative in these tests but pos-itive in the GIFT, the serum is very likely to containgranulocyte-specific antibodies If the tests on lympho-cytes are positive and if it is necessary to ascertainwhether granulocyte-specific antibodies are present,the serum should be absorbed with pooled platelets toremove any HLA class I antibodies or the serum must

be tested with a granulocyte panel typed for specific antigens Unfortunately, positive reactions withlymphocytes particularly in the immunofluorescencetest may be due to lymphocyte-specific antibodies,

granulocyte-in which case it may be difficult to ascertagranulocyte-in the ence of granulocyte-specific antibodies, unless a knownspecificity is detected

pres-With the GIFT, not only antibodies but also formed immune complexes cause positive reactions,

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pre-due to adherence to Fc and complement receptors

(Camussi et al 1979; Engelfriet et al 1984) There are

three possible ways of distinguishing between

anti-bodies and fixed immune complexes:

1 Preparation of an eluate from positively reacting

granulocytes Eluted antibodies will again react with

granulocytes while immune complexes are usually

dis-sociated by the elution procedure (Helmerhorst et al.

1982)

2 Testing the serum under investigation in an ADCC

assay on granulocytes

3 Blocking Fc receptors on target granulocytes with

monoclonal antibodies (Engelfriet et al 1984) In

practice, it is difficult to distinguish between

autoanti-bodies and bound immune complexes, because there

are seldom enough cells to prepare an eluate and

because the results of the ADCC assay on patients’

granulocytes are difficult to interpret

Chemiluminescence test

To prepare suspensions of mononuclear cells and

granulocytes, fresh EDTA blood is centrifuged on

Ficoll-hypaque (density 1.077) The mononuclear

cell fraction is washed three times The red cell/

granulocyte fraction is resuspended in PBS and

mixed 1:4 with dextran solution After sedimentation

(30 min) the granulocytes in the supernatant are

packed and then washed twice in PBS The

granulo-cytes are resuspended in PBS and transferred to a

glass tube pre-located in a waterbath at 52°C and

after 2 min are allowed to cool in another tube at

room temperature The purpose of the heat treatment

is to render the granulocytes incapable of responding

to immunoglobulin aggregates in the sera to be

tested which would lead to non-specific generation of

chemiluminescence

For the assay, granulocytes are incubated with

serum and then washed in phosphate-buffered

solu-tion (PBS) and resuspended in Hanks’ BSS The

gran-ulocytes are then incubated with freshly prepared

mononuclear cells from the peripheral blood and with

luminol Chemiluminescence is generated as a result of

phagocytosis of sensitized granulocytes, which leads

to the formation of oxygen radicals and oxidation of

luminol Chemiluminescence is measured in a

lumino-meter (Hadley and Holburn 1984) The sensitivity of

the chemiluminescence (CL) test is similar to that of

the GIFT (Lucas 1994)

Phenotyping and genotyping of neutrophil antigens

Phenotyping By tradition, neutrophil antigen typing

has been performed using human alloantibodies in thegranulocyte agglutination or GIFT assay However,alloantisera are difficult to obtain Monoclonal anti-bodies specific to HNA-1a, HNA-1b and HNA-2ahave been described and are available commercially.These reagents have been used to phenotype neutro-phils by flow cytometry This method is faster and easier than manual methods with alloantibodies, aswhole blood rather than isolated neutrophils can be used

Genotyping As alloantibodies are difficult to obtain

and monoclonal antibodies are not available for allneutrophil antigens, genotyping assays have gainedimportance Genotyping assays are performed withDNA isolated from whole blood, thus eliminating theneed to isolate granulocytes Furthermore, leucocyteDNA can be stored for months before testing is per-formed The characterization of the genes encodingHNA-1 antigens has led to the development of assays

for these antigens (Bux et al 1995b; Hessner et al.

1996) Genotyping of HNA-1 antigens is particularlyvaluable given the rarity of alloantibodies to HNA-1cand the absence of monoclonal antibodies Geno-typing for FCGR3B alleles is complicated by the high degree of homology between FCGR3B andFCGR3A Among the five nucleotides that differbetween FCGR3A*1 and FCGR3A*2, FCGR3A is thesame as FCGR3A*1 at three nucleotides and the same

as FCGR3A*2 at two nucleotides As a result, mostlaboratories use PCR and sequence-specific primers todistinguish FCGR3A alleles A unique set of primers isused to amplify each of the three alleles

Unfortunately, HNA-2a genotyping reagents are notavailable The HNA-2a-negative phenotype is caused

by CD177 mRNA splicing defects (Kissel et al 2002).

However, no mutations have been detected in theCD177 genomic DNA from subjects with HNA-2a-negative neutrophils It may be possible to distinguishpositive and negative phenotypes by analysing CD177mRNA for accessory sequences, but this is a highlysophisticated methodology

Antigens found only on lymphocytes

In addition to HLA antigens (see above) to some red

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cell antigens (see Chapter 5), 5a and HNA-3a antigens,

and antigens also present on granulocytes and

mono-cytes, lymphocytes carry antigens that do not occur on

other cells

Two biallelic systems, one on Tγ cells with the

antigens TCA1 and TCA2, and one on Tµ cells with

the antigens TCB1 and TCB2 have been defined (van

Leeuwen et al 1982a) Non-HLA antigens only

expressed on activated T cells have been described

(Gerbase et al 1981; Wollman et al 1984) The

clin-ical significance of alloantibodies against

lymphocyte-specific antigens is uncertain, but a case of alloimmune

lymphocytopenia of the newborn, due to maternal

alloantibodies and resulting in severe combined

immune deficiency has been reported (Bastian et al.

1984)

Cold autoantibodies to lymphocytes

Anti-I and anti-i See Chapter 4.

Lymphocyte autoantibodies reactive

at 37°C in vitro

Single cases of (1) hypogammaglobulinaemia with

cytotoxic autoantibodies against B cells reactive at

37°C and (2) acquired hypogammaglobulinaemia

with autoantibodies specific for T-helper cells, leading

to increased activity of T-suppressor cells, have been

described (Tursz et al 1977).

Antibodies against various subsets of lymphocytes

have been described in patients with AIDS and may

contribute to the decline in the CD4+T-cell count

Antigens found only on monocytes

In addition to HLA class I and class II antigens, and

the antigens shared by monocytes and granulocytes

mentioned above, monocytes carry alloantigens that

do not occur on other blood cells Some of these

anti-gens (EM antianti-gens) are also present on endothelial

cells (Moraes and Stastny 1977; Claas et al 1980;

Cerilli et al 1981; Stastny and Nunez 1981); others

are monocyte specific (Cerilli et al 1981; Baldwin et al.

1983; Paul 1984) Antibodies against EM antigens

are detrimental to transplanted kidneys and may be

involved in GvHD EM antibodies and antibodies

react-ing with monocytes, tubular endothelium and kidney

cells in the cortex can be eluted from rejected kidneys

(Joyce et al 1988) The significance of

monocyte-specific alloantibodies needs further evaluation

mole-platelets are absorbed from the plasma (Santoso et al.

1993a) The number of some of the class I antigens onplatelets varies greatly in different subjects Class IIantigens are not detectable on platelets but HLA-DRantigens can be induced at the platelet surface by stimu-lation with cytokines, for example gamma-interferon,

both in vitro and in vivo (Boshkov et al 1992).

Red cell antigens also found on platelets

ABH, Lewis, I, i and P antigens on platelets aredescribed in Chapter 4 Using a sensitive two-stageradioimmunoassay the major antigens of the Rh, Duffy,Kell, Kidd and Lutheran systems have been shown to

be absent from platelets (Dunstan et al 1984).

Antigens found only on platelets (platelet-specific antigens)Several systems have been defined whose antigens arefound on glycoproteins of platelets Some of these anti-gens are found on other cells such as endothelial cells

as well The human platelet antigen (HPA) ture system was adopted in 1990 (von dem Borne andDecary 1990) The HPA nomenclature categorizes allalloantigens expressed on the platelet membrane, exceptthose encoded by genes of the major histocompatibil-ity complex A platelet-specific alloantigen is called aHPA when its molecular basis has been defined Thedifferent HPAs are grouped in systems based on hav-ing alloantibodies defining a given alloantigen and its

nomencla-‘antithetical’ alloantigen A large number of antigenshave been described and the molecular basis of many has been resolved To date, 24 platelet-specificalloantigens have been defined by immune sera, ofwhich 12 are grouped into six biallelic systems (HPA-1,

-2, -3, -4, -5, -15) (Table 13.2 and Metcalfe et al.

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2003) For the remaining 12 antibodies, alloantibodies

against the antithetical antigen have yet to be

discov-ered The molecular basis of 22 out of the 24

serologic-ally defined antigens has been resolved In all but one,

the difference involves a single amino acid substitution

generally caused by a single nucleotide polymorphism

(SNP) in the gene encoding the relevant membrane

gly-coprotein The systems are numbered in the order of

the date of publication and the antigens are designated

alphabetically in the order of their frequency in the

population This nomenclature has been criticized, the

main objection being that HPA-1a, HPA-4a, HPA-6a,

HPA-7a and HPA-8a are five different names for

ident-ical GPIIIa molecules carrying these high-frequency

antigens Only the GPIIIa molecules that carry the

low-frequency antigens of these systems differ from

each other due to amino acid substitutions at different

positions of the molecule (Newman 1994)

HPA-1 system (Zw, Pl A )

The first system to be described was recognized by

van Loghem and co-workers (1959) when a serum was found that agglutinated some samples of plateletsbut not others; the antigen was named Zwawhen anantithetical antigen (Zwb) was recognized (van der

Weerdt et al 1962, 1963) Anti-PlA1(Shulman et al.

1961) was subsequently shown to have the samespecificity as anti-Zwa The system is now namedHPA-1 and the antigens, HPA-1a and HPA-1b.Ninety-eight per cent of white people are HPA-1(a+)and 27% HPA-1 (b+)

The 1 gene has two alleles, 1a and 1b Anti-HPA-1a is associated with most cases of post-transfusion purpura and neonatal alloimmunethrombocytopenia (NATP)

HPA-HPA-1a and -1b antigen sites are situated on themembrane glycoprotein IIIa (Kunicki and Aster 1979;van der Schoot and von dem Borne 1986) The HPA-1polymorphism results from the substitution of a singlebasepair in the coding DNA at position 33, coding for leucine in HPA-1a and for proline in HPA-1b

(Newman et al 1989) Patients with Glanzmann’s

thrombocytopenia type I have no detectable membrane

System Antigen Original names Glycoprotein CD

HPA-4b Yuk a , Pen b

From Metcalfe et al (2003).

Table 13.2 Human platelet antigens

(HPAs).

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glycoprotein IIIa (or IIb) on their platelets and are

therefore unable to express the HPA-1 antigens

(Kunicki et al 1981; van Leeuwen et al 1981).

The HPA-1 polymorphism is not found in Japanese

people (Shibata et al 1986b).

HPA-2 system (Ko)

A second biallelic system, Ko (HPA-2), was described

by van der Weerdt and co-workers (1962) In total,

16% of subjects were found to be HPA-2(b+) (Ko(a+)

and 99% were HPA-2(a+) (Ko(b+) Like anti-HPA-1a,

anti-HPA-2a and -2b were detected by platelet

agglut-ination The HPA-2 antigens are situated on GPIb/IX

(Kuijpers et al 1989) The polymorphism involves

substitution of a single nucleotide in the DNA at

posi-tion 434, which codes for the β-chain of GPIb, to

give methionine in HPA-2b and threonine in HPA-2a

at position 145 (Kuijpers et al 1992a) A platelet

anti-gen Siba, described by Saji and co-workers (1989), was

shown to be identical to Koa(Kuijpers et al 1989).

HPA-3 system (Bak, Lek)

The platelet antigen, Baka(HPA-3a) is present in about

90% of the Dutch population (von dem Borne et al.

1980) The first example of anti-HPA-3a was

respons-ible for NATP An antigen, Leka, at first found to be

closely associated serologically with Baka (Boizard

1984) was subsequently shown to be identical (von

dem Borne and van der Plas-van Dalen CM 1985)

HPA-3a is present on glycoprotein IIb (Kieffer et al.

1984; van der Schoot and von dem Borne 1986) The

antigen HPA-3b, antithetical to HPA-3a, was described

independently by Kickler and co-workers (1988a) and

Kiefel and co-workers (1989a) In both cases,

anti-HPA-3b was responsible for post-transfusion purpura

The HPA-3 polymorphism is also due to the

substitu-tion of a single basepair in the coding DNA, to give

isoleucine at amino acid residue 843 in HPA-3a and

serine in HPA-3b (Lyman and Aster 1990)

HPA-4 system (Pen, Yuk)

Another biallelic system, Yuk (HPA-4) was described

by Shibata and co-workers (1986a,b) Both the

low-frequency antigen Yuka(HPA-4b) and the

high-frequency antigen Yukb(HPA-4a) were detected with

antibodies that caused NATP

The antigen, Pena, that had been described byFriedman and Aster (1985), proved to be identical toYukb(RH Aster and Y Shibata, unpublished observa-

tion) HPA-4a is present on GPIIIa (Furihata et al 1987; Santoso et al 1987) The HPA-4 polymorphism

has not been found in white people (Friedman and

Aster 1985; Kiefel et al 1988) The Yuk

polymor-phism involves substitution of a single nucleotide inthe DNA which encodes the GPIIIa protein, coding forarginine at position 526 in HPA-4a and for glutamine

in HPA-4b (Wang et al 1991).

HPA-5 system (Br, He, Zav, Tu a , Ca a Mo a , Sr a , Max a , La a , Gro a , ly a , Sit a , Oe a )

The antigens Bra (HPA-5b) and Brb (HPA-5a) weredescribed by Kiefel and co-workers (1988, 1989a).The HPA-5 antigens are present on glycoprotein la

(Kiefel et al 1989a; Santoso et al 1989) Anti-HPA-5a

and -5b have been responsible for NATP

Most HPA-5 antibodies are non-reactive in theimmunofluorescence test because of the low number of

antigenic sites (Kiefel et al 1989b) HPA-5 antibodies

can best be detected by the MAIPA, a specific assay (see below) The polymorphism involvessubstitution of a single nucleotide in the cDNA at posi-tion 1648, to give glutamine in HPA-5a and lysine

glycoprotein-in HPA-5b at position 505 (Santoso et al 1993b)

The biallelic Zav system described by Smith and co-workers (1989) is identical with the HPA-5 systemand the antigen Hca is the same as HPA-5b (Woods

et al 1989) Tu/Ca (HPA-6bw), a low-frequency

antigen located on GPIIIa and involved in NATP was

at first named Tua(HPA-6b) (Kekomaki et al 1993)

It is identical to Caadescribed by McFarland and workers (1993) The polymorphism involves a singlenucleotide substitution at position 1564 to give 489glutamine in HPA-6a and 489 arginine in HPA-6b

co-(Wang et al 1993b) The antigen Mo (HPA-7bw),

involved in NATP, is located on GPIIIa and due to

a C–G substitution at position 1267 in the cDNA,resulting in a substitution of proline by alanine at

position 407 (Kuijpers et al 1993) Sra(HPA-8b) has

so far been detected in only one family, in which it wasinvolved in NATP The antigen is located on GPIIIaand is due to the substitution at position 636 of cys-teine (in HPA-8b) for arginine (normally present in

GPAIIIa) (Kroll et al 1990; Santoso et al 1994) Maxa

(HPA-9bw), a low-frequency alloantigen responsible

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for NATP, is located on GPIIb and the polymorphism

is due to a single nucleotide substitution G→A at

posi-tion 2603 (Noris et al 1995) HPA-11bw (Groa) is

located on GPIIIa and involved in NATP It has so

far been found in only a single family A guanine

for adenine mutation was found, predicting an

arginine→histidine substitution at position 633 of the

mature glycoprotein (Simsek et al 1997) The antigen

ly (HPA-12 bw) is a low-frequency antigen located on

the glycoprotein Ib/IX complex Anti-Iyawas the cause

of severe NATP (Kiefel et al 1995) Sita(HPA-13bw),

a low-frequency antigen in the German population,

was identified in a severe case of neonatal alloimmune

thrombocytopenia Sit(a) epitopes reside on platelet

GPIa A threonine→methionine substitution at the

799 position is responsible for formation of the Sit(a)

alloantigen, and diminished platelet aggregation

responses of Sit(a)(+) individuals indicate that the

Thr(799)Met mutation affects the function of the

GPIa–IIa complex (Santoso et al 1999) Oea

(HPA-14bw), a low-frequency alloantigen responsible for a

case of neonatal NATP, has been assigned to platelet

GPIIIa Molecular studies suggest that Oeaarose as a

result of a mutational event from an already mutated

GPIIIa allele (Santoso et al 2002).

HPA-15 system (Gov, Duv)

A biallelic system with the alleles Gova(HPA-15b)

and Govb (HPA-15a) was reported by Kelton and

co-workers (1990) Anti-Govawas found in a patient

with post-transfusion purpura The Gov antigens are

expressed on the CDw 109 protein (Smith et al 1995).

Anti-Duv(a+), directed against an antigen HPA-16bw

(Duva) on glycoprotein GPIIIa has been implicated in

a case of neonatal thrombocytopenia Sequencing of

the exons 2–15 of GPIIIa revealed a single base

sub-stitution 517C→T (complementary DNA) present in a

heterozygous state in DNA from the father leading

to amino acid substitution of threonine for isoleucine

at position within the Arg-Gly-Asp binding domain of

GPIIIa (Jallu et al 2002).

Obsolete systems and systems not yet included

in the HPA nomenclature

DUZO Moulinier (1957), using the antiglobulin

con-sumption technique, demonstrated a platelet antibody

in the serum of a woman whose four children had died

from neonatal purpura The corresponding antigenwas termed ‘DUZO’ However, no second example ofanti-DUZO has been found and this antigen has there-fore become obsolete

PlE system The two alleles of this system (PlE1and

PlE2) were defined by Shulman and co-workers (1964).This system has not been included in the HPA nomen-clature because anti-PlE1was probably an isoantibodyfrom a patient with Bernard–Soulier syndrome andanti-PlE2is no longer available (Shulman 1987)

PlT antigen An antigen, PlT, with a very high quency was described by Beardsley and co-workers(1987) It is present on glycoprotein V

fre-Nak a The antigen Naka is absent in 3 –11% of

Japanese people and is present on GPIV (Ikeda et al.

1989) However, the Nak antigen appears to be a polymorphic determinant of GPIV, Nak(a–) subjectsbeing deficient for GPIV Anti-Nak therefore is not an

non-alloantibody, but an isoantibody (Yamamoto et al.

1990)

Va a The low-frequency antigen Vaa, involved in

NATP, is located on GPIIIa (Kekomaki et al 1992).

Presence of platelet-‘specific’ antigens

on other cells

The antigens of the HPA-1 system are present on

endothelial cells (Leeksma et al 1987; Giltay et al.

1988a) HPA-1a has also been detected on vascular

smooth muscle cells and fibroblasts (Giltay et al.

1988) The HPA-5 antigens are probably also present

on endothelial cells, which express VLA-2

Alloimmunization to platelet antigens

Role of HLA class I antibodies in refractoriness

to platelet transfusions

When no measures are taken to reduce the number

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of leucocytes in red cell or platelet concentrates,

80–100% of patients, depending upon the disease,

may develop HLA antibodies; only 40 –70% patients

treated with immunosuppressive regimens become

immunized (Howard and Perkins 1978; Dutcher et al.

1980, 1981) In the great majority of immunized

patients, the alloantibodies are directed against HLA

class I antigens (Schiffer et al 1976) Primary

immun-ization occurs as early as 10 days after transfusion,

although 3– 4 weeks is more usual; reappearance of

lymphocytotoxic antibodies appeared as early as 4

days in previously sensitized subjects In total, 62% of

women with acute myelocytic leukaemia and previous

pregnancies became alloimmunized following

trans-fusion during cytoreductive therapy (Trial to Reduce

Alloimmunization to Platelets Study Group 1997)

HLA alloimmunization does not necessarily correlate

with the number or schedule of transfusions, at least

when multiple transfusions are administered When

the number of leucocytes in the transfused cell

concen-trate is reduced, the percentage of immunized patients

decreases, and at levels of 1–5 × 106 or fewer, primary

immunization against HLA class I antigens is

pre-vented (see below)

The presence of HLA class I antibodies in a patient’s

serum does not equate with refractoriness to platelet

transfusions The frequency of most HLA antigens

is low and antibodies against them may not react

with the platelets of any randomly chosen donors

or may react with only a few of them Furthermore,

some class I antigens may be expressed so weakly

on the donor platelets that they survive normally,

or nearly normally, in a patient with antibodies

against them Furthermore, patients with

allobodies against HLA class I antiallobodies may form

anti-idiotype antibodies that react with and inactivate

the class I antibodies For this reason refractoriness

to platelet transfusions may be overcome in spite

of continued transfusions of incompatible platelets

(Atlas et al 1993) In a significant percentage of

alloimmunized patients, evidence of alloimmunization

declines or disappears with time (Lee and Schiffer

1987; Murphy et al 1987) Nevertheless, 10 –20%

of patients in large prospective and retrospective

series of thrombocytopenic patients treated for

malignancy become alloimmune refractory

follow-ing transfusion therapy (Trial to Reduce

Alloimmu-nization to Platelets Study Group 1997; Seftel et al.

2004)

Role of platelet-specific antibodies

Even when HLA-matched platelets, either from closerelatives or from random donors, are transfused, 19% of recipients became refractory (Schiffer 1987)

In most cases refractoriness was probably related to HLA incompatibilities that went undetected ratherthan to antibodies to platelet-specific antigens, becausethe latter occur almost exclusively in patients who are strongly immunized to HLA class I antigens Usingthe MAIPA, platelet-specific antibodies were found

in 25% (9 out of 36) of patients with high levels of

HLA immunization (Schnaidt et al 1996) This figure

corresponds well with the observation that fusions of HLA-compatible platelets are unsuccessful

trans-in about 20% of HLA-immunized patients (Saji et al.

antigens (Welsh et al 1977; Claas et al 1981; van Marwijk et al 1991) Formation of such antibodies

occurs if the platelets are contaminated with cytes, but platelets alone are capable of inducing a

leuco-secondary immune response (Gouttefangeas et al.

2000) Although foreign antigen is presented to helper

T cells by the subject’s own HLA class II-positive gen-presenting cells (APCs), the induction of a primaryimmune response to foreign class I antigens must be presented by class II-positive APCs of the donor

anti-(Lechler and Batchelor 1982; Sherwood et al 1986).

Dendritic cells are probably the class II-positive donor

cells responsible for antigen presentation (Deeg et al.

1988) Thus alloimmunization against class I antigensshould be prevented when class II-positive cells havebeen either removed from red cell or platelet concen-trates or inactivated

Removal of leucocytes

An early study showed that patients transfused withplatelet concentrates from which most of the leuco-cytes have been removed, for example by passagethrough cotton wool filters, were substantially lesslikely to become refractory to transfusion of platelets

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(Eernisse and Brand 1981) This observation has

been frequently confirmed (Murphy et al 1986; van

Marwijk et al 1991) A multi-institutional,

random-ized, blinded trial was conducted to determine

whether transfusion of platelets from which leucocytes

had been removed by a filter before storage would

prevent the formation of platelet alloantibodies and

refractoriness to platelet transfusions Patients who

were receiving induction chemotherapy for acute

myeloid leukaemia were randomly assigned to receive

one of four types of platelet transfusions: unmodified,

pooled platelet concentrates from random donors

(control); filtered, pooled platelet concentrates from

random donors (F-PC); ultraviolet B-irradiated,

pooled platelet concentrates from random donors

(UVB-PC); or filtered platelets obtained by apheresis

from single random donors (F-AP) All patients

received transfusions of filtered, leucocyte-reduced red

cells Of 530 patients with no alloantibodies at the trial

initiation, 13% of those in the control group produced

lymphocytotoxic antibodies and their

thrombocytope-nia became refractory to platelet transfusions,

com-pared with 3% in the F-PC group, 5% in the UVB-PC

group and 4% in the F-AP group (P= 0.03 for each

treated group compared with the control subjects)

Lymphocytotoxic antibodies were found in 45% of

the controls, compared with 17–21% in the treated

groups (P< 0.001 for each treated group compared

with the control subjects) Reduction of leucocytes by

filtration and ultraviolet B irradiation of platelets was

equally effective in preventing alloantibody-mediated

refractoriness to platelets during chemotherapy for

acute myeloid leukaemia Platelets obtained by

apheresis from single random donors provided no

additional benefit compared with pooled platelet

concentrates from random donors (Trial to Reduce

Alloimmunization to Platelets Study Group 1997)

Universal pre-storage leucoreduction (ULR) of red

cell and platelet products has been performed in

Canada since August 1999 In a retrospective analysis

of 13 902 platelet transfusions in 617 patients

under-going chemotherapy for acute leukaemia or stem cell

transplantation before (n = 315) and after (n = 302)

the introduction of ULR, alloimmunization was

significantly reduced (19–7%) in the post-ULR group

Alloimmune platelet refractoriness was similarly

reduced (14 – 4%) Fewer patients in the post-ULR

group received HLA-matched platelets (14% vs

5%) Thus leucocyte reduction (see below) reduces

alloimmunization, refractoriness and requirements forHLA-matched platelets when applied as routine trans-fusion practice to patients receiving chemotherapy or

stem cell transplant (Seftel et al 2004) While the total

number of platelet transfusions was also reduced afterULR, this was probably related to other factors such as

a reduction in the platelet transfusion trigger

As might be expected, following the transfusion ofleucocyte-poor platelet concentrates, the development

of platelet refractoriness is much more common inpatients who have been transfused previously or havebeen pregnant, which confirms the suspicion that secondary immune responses to HLA class I antigens

cannot be prevented (Brand et al 1988; Novotny

et al 1995; Sintnicolaas et al 1995) However, in the

two large studies, alloimmunization and alloimmunerefractoriness in patients who were previously preg-nant or transfused were also reduced after ULR (Trial

to Reduce Alloimmunization to Platelets Study Group

1997; Seftel et al 2004).

Counting small numbers of leucocytes

The small numbers of residual leucocytes in filteredconcentrates (1–3 cells/µl) can be determined accur-ately either by flow cytometry, after staining the

nucleated cells with propidium iodide (Wenz et al.

1991), or by using a large-volume counting chamber,

for example the Nageotte chamber (Masse et al.

1991) In trials in 20 laboratories, the detection limitsfor the flow cytometry and NC techniques were 0.1and 1 leucocyte/µl respectively Both methods are suitable for assessing the adequacy of leucodeple-tion filters Sampling error and instrument precisionremain hurdles for all proposed methods Issuesinvolving counting technique and guidelines for pro-cess control of leucoreduced blood components have

been published (Dumont et al 1996; Dzik 2000).

Filters for whole blood and red cell concentrates

There is ample evidence that primary HLA tion does not occur, or occurs only rarely, when fewerthan 5 × 106leucocytes are transfused (Sirchia et al 1982; Saarinen et al 1990; Novotny et al 1995) To

immuniza-avoid primary immunization, the total number of leucocytes transfused in a red cell or platelet concentr-ate must therefore be less than this number For redcells and whole blood collections, the only practical

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