American Journal of Medical Genetics (Semin. Med. Genet.) 106:185±190 (2001)

A R T I C L E

Facilitating Informed Choice in Prenatal Testing:How Well Are We Doing?THERESA M. MARTEAU* AND ELIZABETH DORMANDY

There is a consensus that prenatal testing services need to provide the information and support necessary for womento make informed choices about prenatal testing. Informed choices are those based on relevant information thatre¯ect the decision-maker's values. To date, most research has focused on the information provided to womendeciding whether to undergo tests. This has highlighted the poor quality of information provided to many women.There is agreement on the need to provide information on three key aspects of any test: the condition for which testingis being offered, characteristics of the test, and the implications of testing. Very little research has been conducted ondecisions after the diagnosis of a fetal abnormality and how information and emotional and decisional support are andshould be provided. Research is now needed in four key areas: ®rst, on the optimal ways of organizing services tofacilitate choices that are not only based on relevant information, but also re¯ect the decision-maker's values; second,on the most effective ways of framing information needed for the different decisions involved in prenatal testing; third,on the most effective media in which to deliver information; and, fourth, to identify aspects of counseling that facilitateinformed choices following diagnoses of fetal abnormality. If we value women's ability to make informed choicesabout prenatal tests as highly as we value reliable laboratory tests, evidence-based quality standards need to bedeveloped for the information and support women are given at all stages of the process of prenatal testing.ß 2001 Wiley-Liss, Inc.

KEY WORDS: prenatal testing; informed choice; knowledge; communication of risk information

INTRODUCTION

The past decade has been characterized

by a huge increase in prenatal testing.

There has been a somewhat slower

realization that the information many

women receive about such tests is poor.

This article reviews the evidence con-

cerning the gap between existing guide-

lines and practice and considers how, in

the light of available evidence, practice

can be improved and what evidence is

needed to develop further, evidence-

based practice.

INFORMED CHOICES

There is a strong consensus in ethical

guidelines published in Europe, the US

and elsewhere that health professionals

providing prenatal testing services

should give women and their partners

the information and support they need

to make autonomous, informed deci-

sions [RCP, 1989; Andrews et al., 1994;

Baumiller et al., 1995]. As well as health

professionals and policy makers, users of

prenatal services also place a high value

on services that allow them to make

an informed choice about prenatal

tests [Dodds and Newburn, 1997; Car-

roll et al., 2000]. Such an emphasis

re¯ects a commitment to distance

the practice of current genetics from

past abuses, particularly those in Nazi

Germany [Duster, 1990; Muller-Hill,

1988]. It also re¯ects a growing re-

cognition that it is unethical for in-

dividuals not to be informed of the

There is a strong consensus

in ethical guidelines

published in Europe, the

US and elsewhere that

health professionals

providing prenatal testing

services should give women

and their partners the

information and support

they need to make

autonomous,

informed decisions.

consequences of medical interventions,

particularly ones in which risk informa-

tion is being provided [Kenen, 1996].

Theresa Marteau (PhD, CPsychol) is Pro-fessor of Health Psychology and Director ofthe Psychology and Genetics Research Groupat King's College, London. Over the past20 years she has been conducting researchon psychological aspects of prenatal testingand other types of health risk assessment.

Elizabeth Dormandy (MSc) is ResearchFellow in the Psychology and GeneticsResearch Group at King's College, London.Trained in biochemistry, she worked forthree years educating health professionalsabout prenatal screening. She is currentlycompleting the ®nal year of her PhDexamining the in¯uence of health profes-sionals and service delivery upon uptake ofprenatal screening tests.

Grant sponsor: The Wellcome Trust.*Correspondence to: Theresa M.

Marteau, Psychology and Genetics ResearchGroup, King's College, London, Thomas GuyHouse, Guy's Campus, London SE1 9RT, UK.E-mail: theresa.marteau@kcl.ac.uk

DOI 10.1002/ajmg.10006

ß 2001 Wiley-Liss, Inc.

There is also a belief that an informed

choice, compared with an uninformed

one, is associated with better patient

outcomes. Few studies, however, have

examined the psychological conse-

quences of informed choices in relation

to prenatal or other types of tests. In

other areas of health care, however, there

is good evidence that psychological

preparation for stressful medical proce-

dures is associated with better psycholo-

gical and clinical outcomes [ Johnston

and Vogele, 1993]. How much bene®t

informed choices, as opposed to choices

that are uninformed, confer in those

declining or those undergoing prenatal

testing remains to be determined.

Attempts to quantify such bene®ts may

provide greater incentives than currently

exist to provide services that facilitate

informed choices.

Despite a consensus on the impor-

tance of informed choice there are few

de®nitions and even fewer measures.

There is, however, an emerging con-

sensus that an informed choice or

decision has two core characteristics: it

is based on relevant, good quality

information, and re¯ects the decision-

maker's values [Marteau et al., 2001].

Most research to date has focused

on information provision and under-

standing as indicators of the extent

to which decisions are informed. Very

little research has focused on women's

values to determine, ®rst, the extent to

which their decisions re¯ect their values,

and, second, how services are best

organized to allow choices to re¯ect

values.

INFORMING WOMEN

Women value high-quality information,

given personally by health professionals

and as early as possible in prenatal care to

allow time for re¯ection and informed

decision-making [Dodds and Newburn,

1997; Carroll et al., 2000]. Unfortu-

nately, the experiences of many women

fall short of this. Many studies, for

example, document women's poor levels

of knowledge about the tests they are

offered [Marteau et al., 1988; Smith

et al., 1994a; Chilaka et al., 2001]. A

professional consensus is evident in

several sets of guidelines concerning

the information that women need when

offered prenatal testing [American Col-

lege of Obstetricians and Gynecologists,

1987; Joint Working Party of the Royal

College of Obstetricians and Gynae-

cologists and the Royal College of

Paediatric and Child Health, 1997].

These include 1) information on the

condition for which testing is being

offered, 2) the characteristics of the test,

and 3) the implications of possible test

results.

Information that women

need when offered prenatal

testing include 1) information

on the condition for which

testing is being offered,

2) the characteristics of the

test, and 3) the implications

of possible test results.

Information About

the Condition

The information provided about the

conditions for which testing is provided

tends to be brief [Murray et al., 2001;

Marteau et al., 1992]. In an overview of

information presented about prenatally

diagnosed conditions, Asch [1999] con-

cluded that the information provided

was too negative, given the results of

surveys showing that disability most

often does not lead to an unsatisfying

life. Information provided on conditions

is generally more negative when pro-

vided to those considering prenatal

testing than to those considering testing

at other times or to parents with an

affected child [Lippman and Wilfond,

1992; Loeben et al., 1998]. This raises

the question of what comprises a

balanced picture of life with a disability.

Those with a disability perceive the

conditions as less serious than those

without it [Marteau and Johnston,

1986]. Thus, adults with a condition

are less likely to endorse termination of

pregnancies affected by their condition

than are parents with affected children

[Conway et al., 1994; Henneman et al.,

2001]. Whatever the origins of the

differences, their existence indicates the

importance of eliciting the views of

many groups in order to obtain as full a

view as possible of how to present

conditions to prospective parents. The

need to assess the impact upon decision-

making of different views is suggested by

the counterintuitive results of an analog

study, comparing the effects of different

pictures of children with Down syn-

drome [Figueiras et al., 1999]. A picture

portraying a positive image of a child

with Down syndrome had a similar

impact as a negative image: both resulted

in higher concerns about having a child

with the condition, compared with the

level of concern generated in' those

given no picture. Prospective studies are

needed to compare outcomes for parents

making different decisions based on

different types and amounts of informa-

tion about conditions.

Characteristics of the Test

Most people have unrealistic expecta-

tions of screening tests, overestimating

the number of cases they can detect and

underestimating the number of people

recalled who are subsequently shown to

have no problems [Cockburn et al.,

1995]. Few studies have compared the

different ways in which such informa-

tion can be presented. There is good

evidence from other areas to show that

quantifying uncertainty using frequen-

cies (e.g., 10 in every 1,000 women)

rather than numerical probabilities (e.g.,

1 in 100 chance) leads to greater under-

standing [Gigerenzer and Hoffrage,

1995; Hoffrage and Gigerenzer, 1998].

Two studies in the area of prenatal

testing evaluating women's understand-

ing of the risks following screening for

Down syndrome show better under-

standing when risks are presented using

numbers rather than words [Marteau

et al., 2000] and, when numbers are

presented, using frequencies rather than

probabilities [Grimes and Snively, 1999].

More research of this type is needed,

given the centrality of risk perceptions

186 AMERICAN JOURNAL OF MEDICAL GENETICS (SEMIN. MED. GENET.) ARTICLE

to women's decision-making about pre-

natal tests [Markens et al., 1999].

Implications of Testing

To make an informed choice about

prenatal tests, women need information

on the outcomes of testing. These in-

clude the possibility of a miscarriage

following invasive diagnostic tests as well

as the options available following the

diagnosis of a fetal abnormality. For

diagnostic tests, it is important to convey

the risks inherent in such procedures.

The possibility of a miscarriage can be

presented in numerous ways. Results

from a recent analog study suggest that

the framing of such risks might affect

women's decisions [Shiloh et al., in

press]. Further work is needed to

ascertain, ®rst, the extent to which such

variations in¯uence actual decisions,

and, second, how ways of framing the

information result in optimal levels of

informed choice. The options following

the diagnosis of a fetal abnormality

include termination of the affected preg-

nancy as well as continuing with the

pregnancy, while preparing for the birth

of a child with special needs. Informa-

tion on the ®rst option as opposed to the

second option is more often given,

although it is not always provided either

in writing or orally [Loeben et al., 1998;

Marteau et al., 1994; Bernhardt et al.,

1998]. Information is less often given

about the impact of living with a child

with a disability, including the educa-

tional and medical support that is avail-

able, and the likely impact of such a

choice upon family life. Evidence-based

information that prospective parents

would ®nd useful is most likely that

Evidence-based information

that prospective parents

would ®nd useful is most

likely that which is gathered

from multiple sources.

which is gathered from multiple sources

[Holmes-Rovner et al., 2001].

MEDIA FOR PROVIDINGINFORMATION

Information can be presented using

one or more media. Most often it is

presented in writing and orally, by

health professionals providing prenatal

care.

Written Information

Written information about prenatal tests

varies widely in length, areas covered,

and the ways in which the information is

framed. For example, Murray et al.

[2001] examined the extent to which

81 lea¯ets used in British screening

programs met criteria for good quality

lea¯ets. Overall, the quality of the

lea¯ets was judged to be poor. In terms

of factual content, only 11 (14%)

included information on all eight items

recommended by a professional body

[RCOG, 1993]. In a quantitative analy-

sis of 28 lea¯ets produced for US and UK

cystic ®brosis carrier testing screening

programs, the amount of information

provided ranged from 1±37 statements

(median� 6.5), with most statements

being classi®ed as neutral and only a

minority conveying a positive or nega-

tive image of the condition [Loeben

et al., 1998]. Positive statements were less

common in UK lea¯ets, commercially

produced lea¯ets, and lea¯ets describing

prenatal carrier testing. The impact of

such variations on uptake of tests has not

been described. Evidence suggesting

that the impact of lea¯ets is likely to

be quite small, compared with the

impact of orally presented information

or the way services are delivered,

comes from a study in which uptake

of prenatal screening in 12 hospitals

using the same lea¯et was more varied

than uptake across nine hospitals using

different lea¯ets [Dormandy et al., in

press].

Orally Presented Information

Observational studies of health profes-

sionals presenting prenatal tests to

women attest to the complexities facing

health professionals in communicating

with women with diverse needs [Rapp,

1987]. Interview studies with women

suggest that their attitudes toward

undergoing tests are shaped in various

ways by these health professionals [Press

and Browner, 1997].

Tape recordings of consultations

both in the US and the UK in which

prenatal screening tests are being pre-

sented reveal that the information pro-

vided is insuf®cient for informed

decision-making, providing too little

information, which is occasionally mis-

leading or inaccurate [Marteau et al.,

1992; Bernhardt et al., 1998]. Several

factors could explain this. First, health

professionals themselves sometimes

do not have suf®cient knowledge

about the tests [Smith et al., 1994b;

Sadler, 1997]. Second, they can lack

even basic counseling skills needed to

present information in a way that

makes it understandable [Smith et al.,

1995]. Third, they may lack the time

to present the information [Green,

1994]. More generally, the lack of

high-quality information provided at

many centers may re¯ect negative atti-

tudes toward providing such informa-

tion [Green, 1994; Khalid et al., 1994].

Understanding more about such atti-

tudes could prove an important step in

increasing the quality of information

women receive from their health care

providers.

Brief training can improve both

health professionals' knowledge and

their skills at providing information

[Smith et al., 1995]. However, participa-

tion in this trial was low, with only 69%

of midwives and obstetricians appro-

ached to participate in the study accept-

ing, with those accepting holding

more positive attitudes toward such

training than those who declined. Of

those who did accept, only 40% com-

pleted the training and evaluation. Of

more concern is the fact that those

who completed the trial, compared

with those who dropped out, had

better baseline communication skills

and provided more information to

patients, suggesting that those most in

need of training are those less likely

to avail themselves of it [Michie and

Marteau, 1999]. This suggests the

need for mandatory training programs.

ARTICLE AMERICAN JOURNAL OF MEDICAL GENETICS (SEMIN. MED. GENET.) 187

In addition, such training should form an

integral part of medical and nursing

training, as well as the training of genetic

counselors.

Other Media for

Communicating Information

Two studies evaluated the effectiveness

of videos in providing information.

One found an increase in knowledge

[Hewison et al., 2001]; the other did

not [Michie et al., 1997]. There is

increasing interest in the use of

more interactive media. While interac-

tive media have been found to be

effective in facilitating informed deci-

sion-making for other screening tests

[O'Connor et al., 1999], the one trial of

their use for prenatal screening did not

®nd an advantage over a well-written

lea¯et [Graham et al., 2000]. As Web-

based decision aids continue to develop

it seems likely that the provision of

information tailored to a woman's needs

will prove a sensitive and effective way of

informing her. Mindful of the ``digital

divide,'' it will be important that such

facilities are available in health care

settings to ensure that health dis-

crepancies are not enhanced further by

making high-quality information avail-

able using media unavailable to poorer

women.

SERVICE DELIVERY

Given that an informed choice is one

that re¯ects a woman's own values,

research is needed to determine the ways

of presenting tests that enable choice to

re¯ect a woman's values. For example,

more women undergo tests when they

are presented as part of a routine visit,

not requiring a separate visit [Lorenz

et al., 1985; Bekker et al., 1993;

Dormandy et al., in press; Tambor

et al., 1994]. It is unclear which system

of offering tests results in choices that

most re¯ect women's values. Other

aspects of service delivery that may affect

the extent to which choices women

make are informed is the amount of time

health professionals have to present these

choices. Only by using a valid measure of

informed choice that assesses both

knowledge and women's values [e.g.,

Marteau et al., 2001] can we ascertain

the extent to which different character-

istics of service delivery impede or faci-

litate the making of an informed choice.

Given the very large effect service deli-

very can have on use of testing [Bekker

et al., 1993; Tambor et al., 1994], this

research question should assume some

urgency.

DECISIONS AFTER THEDIAGNOSIS OF A FETALABNORMALITY

In contrast to the great volume of

research on women's decisions about

prenatal testing, there is a dearth of

research on women's decisions following

the diagnosis of a fetal abnormality.

Consequently, little is known about

how best to facilitate women's decision-

making after the diagnosis of a fetal

abnormality. There are many barriers to

conducting research on this. The num-

bers of women affected are small,

making large, multicentered studies a

requirement to ask even the most simple,

descriptive questions. The diagnosis of a

fetal abnormality is invariably a distres-

sing event for women and their provi-

ders, making it dif®cult to seek consent

to participate in research. Creative ways

are needed of overcoming these and

other barriers to research aimed at

determining how best to facilitate

informed choices after the diagnosis of

a fetal abnormality. Research approaches

that merit attention include the use of

simulated patients, to study health pro-

fessionals' counseling, as well as pro-

spective single case studies, to study

long-term adjustment to the decisions

made.

Both the information women

receive as well as the emotional and

decisional support provided are impor-

tant [Biesecker, 2001]. The results from a

small study, in which health professionals

described the information they had

recently given parents following the

diagnosis of a sex chromosome anomaly,

suggest that some parents are being given

inadequate and incorrect information

on which to base their decisions about

whether to continue with the affected

pregnancy [Abramsky et al., 2001]. The

absence of other studies in this area make

it dif®cult to estimate the scale of this

problem.

As well as giving accurate informa-

tion, it is important that health profes-

sionals provide counseling that facilitates

informed choices without being direc-

tive. While the great majority of women

in a recent study reported not being

in¯uenced by health professionals in

deciding whether or not to continue

with an affected pregnancy [Statham and

Solmou, 1998], ®ndings from several

observational studies suggest otherwise.

Three studies show parents are more

likely to terminate pregnancies affected

by a sex chromosome anomaly when

counseled by an obstetrician than by a

geneticist or a specialist pediatrician

[Holmes-Siedle et al., 1987; Robinson

et al., 1989] (Marteau et al., submitted).

Such®ndings re¯ect evidence from seve-

ral other studies suggesting that the

counseling provided by geneticists tends

tobemorepositive and less directive than

that provided by other health profes-

Thirty years after the routine

introduction of prenatal

diagnostic tests, we remain

unaware of how women are

counseled, the information

and support they receive, and

how this affects the quality

and type of decisions

they make.

sionals [Geller et al., 1993; Marteau et al.,

1994]. It is possible that the in¯uence of

health professionals on decisions after

the diagnosis of a fetal abnormality varies

across conditions, exerting a stronger

in¯uence with less familiar and less

severe conditions. While these cross-

sectional data suggest counseling may be

in¯uencing decisions, prospective study

designs are needed to con®rm this. Such

studies need to identify aspects of coun-

seling that facilitate and those that

188 AMERICAN JOURNAL OF MEDICAL GENETICS (SEMIN. MED. GENET.) ARTICLE

impede informed choices following the

diagnosis of a fetal abnormality. While

there is a consensus that directive coun-

seling is undesirable, there is less con-

sensus on what constitutes the elements

of counseling that are desirable; that is,

facilitate informed choices.

Until such time as there is evidence

concerning the most effective ways of

presenting information and providing

decisional and emotional support to

facilitate informed choices, it may be

useful for guidelines to be generated,

using consensus development methods

[Black et al., 1999]. Alongside health

professionals, it would be important

that participants include parents of

affected children and parents who opted

to terminate an affected pregnancy. It

would also be important to include,

where possible, adults with the condi-

tion, given the differences between

their views and those of parents with

affected children [Conway et al., 1994;

Henneman et al., 2001]. The output of

such a process would provide an impor-

tant framework within which counse-

lors could operate. It should not,

however, be construed as a straitjacket

or a unitary model for counseling. The

strength of all counseling lies in the

ability of the counselor to establish a

warm, empathic relationship within

which the needs of the counselee take

center stage.

CONCLUDING COMMENTS

There is now good evidence attesting to

the variable and sometimes poor infor-

mation and support given to women

offered and undergoing prenatal tests.

Enough is known to set basic quality

standards that all centers offering pre-

natal testing should reach. Research now

needs to focus on evaluating different

ways of presenting the same information

and the effectiveness of using different

media and methods of delivering ser-

vices. Outcomes need to expand beyond

knowledge to incorporate an assessment

of women's values to allow evaluation of

the extent towhich different ways of pre-

senting information and tests allow

informed choices to be realized. Evi-

dence is also needed on the size of the

psychological bene®ts of choices that are

informed.

There has been little research

addressing the extent to which services

meet the needs of the few women in

whom a fetal abnormality is diagnosed.

Thirty years after the routine introduc-

tion of prenatal diagnostic tests, we

remain unaware of how women are

counseled, the information and support

they receive, and how this affects

the quality and type of decisions

they make. There is an urgent need to

®ll this gap as a ®rst step towards

providing the counseling known to

facilitate the best outcomes for women

facing such dif®cult decisions.

Laboratories are required to adhere

to quality standards in the conduct of

their tests. No such required quality

standards exist for the provision of

information about such tests. If we value

women's ability to make informed

choices about prenatal tests as highly as

we value reliable laboratory tests, evi-

dence-based quality standards need to be

developed for the information women

are given at all stages of the process of

If we value women's ability

to make informed choices

about prenatal tests as highly

as we value reliable laboratory

tests, evidence-based quality

standards need to be developed

for the information women

are given at all stages of

the process of prenatal

testing.

prenatal testing. This should be a long-

term objective for this area.

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