Antenatal thalassaemia carrier testing: women's perceptions of "information" and "consent".

Abstract

To explore the attitudes of a sample of pregnant women in the UK towards informed consent for antenatal thalassaemia carrier testing and perceived pre-test information needs for such testing.
The study was conducted in two cities in the North of England, where participants were recruited via Midwifery and Genetic services.
In all, 110 Pakistani women tested and not found to be thalassaemia carriers completed a questionnaire, 14 of whom were also interviewed. Thirty-six women identified as carriers or possible carriers completed a questionnaire and were interviewed. The questionnaires assessed whether women were aware that they had been tested for thalassaemia carrier status, whether they were asked for their consent for such testing, and their pre-test information preferences. The interviews explored women's beliefs about "informed consent" in more depth.
Women had received little or no pre-test information and said that they would have preferred to be informed that they were being tested, but they did not expect, or express a desire, to be asked for their informed consent.
While information was important to women, consenting was not. Overall, women discussed "information" and "consent" as two separate issues, thus challenging assumptions around the term informed consent. Women wanted pre-test information because they wanted to know more about the tests that they would be having, not to use it to make decisions about whether to have the tests.

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ORIGINAL ARTICLE
Antenatal thalassaemia carrier testing: women’s
perceptions of ‘information’ and ‘consent’
ShenazAhmed, JosephineGreen, JennyHewison
..................................................................................................
J Med Screen 2005;12:69–77
See end of article for
authors’ affiliations
...............
Correspondence to:
S Ahmed, Academic Unit
of Psychiatry & Behavioural
Sciences, University of
Leeds, Leeds LS2 9LT, UK;
s.ahmed@leeds.ac.uk
Accepted for publication
3 March 2005
Objectives: To explore the attitudes of a sample of pregnant women in the UK towards informed
consent for antenatal thalassaemia carrier testing and perceived pre-test information needs for such
testing.
Setting: The study was conducted in two cities in the North of England, where participants were
recruited via Midwifery and Genetic services.
Method: In all, 110 Pakistani women tested and not found to be thalassaemia carriers completed a
questionnaire, 14 of whom were also interviewed. Thirty-six women identified as carriers or possible
carriers completed a questionnaire and were interviewed. The questionnaires assessed whether
women were aware that they had been tested for thalassaemia carrier status, whether they were asked
for their consent for such testing, and their pre-test information preferences. The interviews explored
women’s beliefs about ‘informed consent’ in more depth.
Results: Women had received little or no pre-test information and said that they would have preferred
to be informed that they were being tested, but they did not expect, or express a desire, to be asked for
their informed consent.
Conclusion: While information was important to women, consenting was not. Overall, women
discussed ‘information’ and ‘consent’ as two separate issues, thus challenging assumptions around the
term informed consent. Women wanted pre-test information because they wanted to know more about
the tests that they would be having, not to use it to make decisions about whether to have the tests.
INTRODUCTION
F
Committee believes that screening programmes should offer
the choice of screening tests to individuals.1Guidelines
recommend that ‘adequately informed consent should be a
requirement for all genetic screening programmes’.2,3
There is no universal definition of ‘informed consent’,
although there is general agreement in the literature that it
has two core characteristics: firstly, that the decision to
accept or decline the test is based on good-quality informa-
tion; and secondly, that it is consistent with the decision-
maker’s values.4,5There is also little agreement on the
amount of pre-test information that should be provided.
Suggestions for pre-test information include the follow-
ing:2,6–8
ollowing recognition of the potential adverse psycho-
logical consequences after receipt of test results and
changing social attitudes, the National Screening
?
?
?
?
?
?
?
the purpose of the screening test;
a description of the genetic condition, including its
severity, variability and therapeutic options;
the genetic inheritance pattern of the condition;
the reliability of the screening test, including false
positives and false negatives;
the procedure for informing individuals of both negative
and positive results;
the implications of a positive result for their future and
existing children, and for other family members;
an explanation of the subsequent options, such as
testing partner, prenatal diagnosis (chorionic villus
sampling or amniocentesis – both have 1–2% risk of
miscarriage), termination/continuation with pregnancy;
?
?
?
a warning for pregnant women that genetic screening
tests may reveal information about paternity;
the uncertainties and risks attached to the screening
process;
any significant medical, social or financial implications
of screening tests.
The literature also suggests that this information should
be presented in a neutral, non-directive manner, allowing
individuals to assimilate the information in accordance with
their own values.6,7In addition, ‘adequately informed
consent should be a requirement for all genetic screening
programmes and to help people decide whether or not to be
screened, it is important to provide both written and oral
information in a language appropriate to the individual’.2
Thalassaemia
b-thalassaemia major (thalassaemia) is a serious recessive
genetic condition that is prevalent among people of
Mediterranean, Middle Eastern and South Asian (Indian,
Pakistani, Bangladeshi) origin. Individuals with thalassae-
mia are unable to make sufficient and sustainable adult
haemoglobin (Hb), so they require regular monthly blood
transfusions. This unfortunately results in excess iron,
which has to be removed daily by injecting a drug called
Desferal slowly under the skin for 8–12h using a pump-
driven syringe.9,10Generally, thalassaemia is not a curable
condition, although bone marrow transplantation offers the
possibility of cure to those who have an immunologically
matched sibling.11The birth of a child with thalassaemia can
be prevented if ‘at-risk’ couples opt for prenatal diagnosis
69
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and subsequently opt for termination of an affected fetus.
At-risk couples are those where both parents are carriers of
the faulty gene, and, on average, one in four of their
pregnancies will be affected. Ideally, prospective parents
from the Mediterranean, Middle East and South Asia would
know before pregnancy whether or not they were carriers,
but in practice this is generally not the case. Therefore, at
the time of this study, selective antenatal thalassaemia
carrier testing was being carried out in many parts of the
UK, where local policies stated that midwives should
routinely offer testing for Hb abnormalities to all pregnant
women who are not of Northern European origin. If a
woman was found to be a carrier, then her partner would
also be offered carrier testing; she would not be at risk of
having a child with thalassaemia unless both parents were
carriers.
Thalassaemia carrier testing in practice
In order to understand how the various test results are
derived and how possible issues about informed consent can
arise, it is important to look at the laboratory diagnosis of
thalassaemia carriers, which leads to the diagnosis of
a-thalassaemia and b-thalassaemia carriers, and of other
Hb variants. This process involves three stages (Figure 1).
Stage 1
This involves measuring the mean cell Hb level (MCH),
where an MCH less than 27 per gram (pg) indicates the
presence of a thalassaemia (a or b), iron deficiency, or both.
If the MCH is less than 27pg, further investigations are
carried out to identify b-thalassaemia carriers (i.e. Stage 2).
It is important to note that this first measure for identifying
thalassaemia carriers forms part of routine antenatal blood
investigations and further testing only takes place if this
routine measure indicates an abnormality. This then means
that all pregnant women are in fact screened for thalassae-
mia carrier status.13So, in selective antenatal testing,
women of Northern European origin to whom testing has
not been offered may nonetheless be tested for thalassaemia
carrier status (Stages 2 and 3 in Figure 1), without having
given prior consent, if they have an MCH less than 27pg.
Guidelines do not specify how to obtain consent for
antenatal testing programmes where the initial indicator of
an ‘abnormality’ is a routine measure.
Stage 2
Investigations at this stage can lead either to the diagnosis of
b-thalassaemia carriers or to inconclusive results which
require further investigations. Stage 2 involves measuring
the HbA2 level, where a value over 3.5% confirms
b-thalassaemia carrier status. However, inconclusive results
are produced at this stage if the HbA2level is less than or
equal to 3.5%, indicating the presence of iron deficiency,
a-thalassaemia, or rare forms of b-thalassaemia, which can
only be diagnosed following DNA analysis. Given that
individuals with inconclusive results could be thalassaemia
carriers, they are referred to from here onwards as ‘possible
carriers’.
Stage 3
This stage involves further investigations, including DNA
analysis, which can take four to six weeks, so the expedient
option is to assume that ‘possible carriers’ are in fact
thalassaemia carriers and employ the same process for
subsequent actions as for b-thalassaemia (i.e. offer carrier
testing to their partners –even though these possible carriers
are likely to be a-plus thalassaemia carriers, which has little
clinical significance, or simply iron deficient).
At the NHS Trusts where the study was conducted, the
midwives were operating the agreed selective testing
protocol and understood that only women who were not
of Northern European origin should be offered or informed
about thalassaemia carrier testing, and believed that only a
request from them would trigger the laboratory to perform
such testing.14They were unaware that the MCH measure,
routinely performed on all pregnant women for other
purposes, formed the first stage of such testing, and that
an abnormal MCH measure would automatically lead to
further laboratory investigations for thalassaemia carrier
status.
Red cell indices (STAGE 1)
MCH < 27pg – could be thalassaemia
or iron deficiency
Hb A2 = or < 3.5%
Hb A2 estimation (STAGE 2)
? Iron deficiency
? α−thalassaemia
? Carrier of rare form of
β−thalassaemia
(STAGE 3)
Further investigations
including inspection of
indices and DNA
analysis if necessary
Hb A2 > 3.5%
MCH > 27pg
β−thalassaemia carrier
No thalassaemia
Figure 1
The laboratory diagnosis of thalassaemia carriers (adapted from Modell and Anionwu12)
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Assumptions associatedwith informedconsent
There are a number of assumptions associated with the
concept of informed consent. These include: people wish to
be informed; people wish to make choices about their care;
and people wish their consent to be sought. Literature on
desire for information and desire to make decisions indicates
that while people want more information on whatever
medical procedure they are about to undergo, many people
prefer health professionals to make decisions on their
behalf.15,16Providing choices could result in women being
faced with complex decisions. In addition, people are
unlikely to become autonomous decision-makers and it
could be considered unethical to force people to make
decisions they may not want to make.15Nevertheless, bio-
ethicists argue that people have a duty to be informed, to
learn about themselves and their potential fate.17Rather
than arguing about whether health professionals or patients
should make decisions about genetic testing, others suggest
that making such a decision in a medical context lies on a
continuum and that the aim should be for mutual decision-
making, or at least the patients’ values being taken into
account before making a decision on their behalf.18In
addition, there is no evidence to show whether people from
different communities share informed consent as a value. It
may be that some people place greater emphasis on health
professionals’ judgements (i.e. ‘They [health professionals]
know best’)19and prefer to trust health professionals to
make health-related choices for them.20
Information is considered a prerequisite for antenatal
testing for genetic conditions, but there is little guidance as
to the content of pre-test information. Few studies have
looked at people’s views about informed consent; their
perceived pre-test information needs for antenatal testing
for genetic conditions; and whether and how people use
pre-test information to consent to such testing. This paper
describes the attitudes of a sample of pregnant women
towards informed consent for antenatal thalassaemia carrier
testing and their perceived pre-test information needs for
such testing.
METHOD
Setting
The study was conducted in two cities in the North of
England. Potential participants were accessed though Mid-
wifery and Genetic services.
Design
The study included three groups of pregnant women who
had been tested for thalassaemia carrier status and diag-
nosed as either b-thalassaemia carriers, possible thalass-
aemia carriers or not thalassaemia carriers. The original plan
was to focus on Pakistani women because the hospitals
involved in the study explained that they had a selective
antenatal testing policy. However, on discovering that
testing was universal in practice, a decision was taken to
include all pregnant carriers and possible carriers of any
ethnic origin.
Recruitment procedures
Approval was obtained from the relevant Local Research
Ethics Committees. All carriers and possible carriers were
recruited through thalassaemia counsellors, usually after
they had received their partner’s carrier test results. In City
A, 26 women were identified as carriers over a period of 18
months (September 1999–March 2001). Twenty-two of
these carriers were Pakistani, 15 of whom agreed to take
part in the study. In City B, both pregnant Pakistani and
white indigenous women were identified as thalassaemia
carriers. Over a period of six months (1 August 2000–31
January 2001), 17 women were identified as b-thalassaemia
carriers. Ten of these carriers were not approached to take
part in the study either because they were not Pakistani or
white indigenous women, or because their partner had not
yet been tested (so had not been through the whole testing
process). Four of the seven carriers approached agreed to
take part in the study. Seventeen of the 36 possible carriers
approached took part in the study: 15 of 23 asked on the
telephone by the Antenatal Screening Coordinator and 2 of
13 who were asked by letter.
Over a period of eight months (February–September
2000), non-carriers were approached to take part in the
study (initially by their midwife) after their second antenatal
clinic visit, which is when they were provided with the
results of their antenatal tests, including thalassaemia carrier
status. At this stage, women should, in theory, have known
which tests they had had and the results of these tests.
Initially, the midwife briefly told the women about the study
and introduced them to the researcher. Of the 125 non-
carrier women who were given details about the study by
the researcher, 110 agreed to take part in the study.
Data collection
All the women completed a questionnaire and all the
carriers, possible carriers and 14 of the non-carriers were
interviewed. The questionnaire asked pregnant women
whether they were told and whether they were asked
about thalassaemia carrier testing when their blood was
taken for testing, and whether they would have wanted to
know that they would be tested. Women were offered the
choice of whether they wanted to complete the question-
naire in English or Urdu, and whether they wanted to
complete the questionnaire themselves or with the aid of
the researcher.
The researcher conducted all interviews in women’s
homes and in their chosen language (English, Urdu,
Punjabi, Mirpuri or Hindu), using a guide developed from
a review of the existing literature. Questions relating to
informed consent explored:
?
women’s perceived pre-test information needs, includ-
ing their reasons for their pre-test information prefer-
ences, the type, amount and timing of information
preferred, and barriers to acquiring information;
women’s attitudes toward consent for antenatal thalas-
saemia carrier testing.
?
The interviews lasted between 30 and 60min and were
audiotaped with permission. Interviews were then trans-
lated where necessary and transcribed by the first author.
Non-carriers completed the questionnaire either in the clinic
immediately after their consultation with the midwife, or at
home.
Analysis
The grounded theory approach was used for qualitative data
collection and analysis, supported by N-Vivo (Nudist-Vivo
1.2; SAGE Publications). Interviews and data analysis were
Perceptions of ‘information’ and ‘consent’ 71
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conducted simultaneously to allow for exploration of
emergingthemesinsubsequent
showed that saturation was reached. The first author
analysed the transcripts using the constant comparative
method.21This involved coding data line by line to identify
and assign codes to meaningful words, phrases or sentences.
Codes were also given descriptions to allow comparison
between them. Axial coding was carried out in parallel to
open coding, which resulted in clustering codes with similar
concepts into categories. The clustering of categories
produced higher-order categories, which in turn were
combined to develop themes to explain phenomena.
The findings for the three groups are presented together to
provide an in-depth explanation of phenomena; that is, by
including the perspective of pregnant women with different
outcomes following antenatal thalassaemia carrier testing.
The quantitative and qualitative findings are also presented
together. Most of the findings in the Results section have
been derived from the qualitative analysis. The quantitative
interviews.Analysis
results consist of percentages and frequencies and are
presented at the beginning of subsections. All names used
in the Results and Discussion sections are pseudonyms.
RESULTS
Sample
The participants were 146 pregnant women tested for
thalassaemia carrier status (see Table 1 for participants’
demographic details). Nineteen of these had been diagnosed
as carriers, 17 as possible carriers and 110 as non-carriers.
During the interviews, questions were asked separately
about information and consent, which is reflected in the
presentation of the results.
Provision of pre-test information for thalassaemia
carrier testing
The researcher told women that the routine antenatal blood
tests included testing for thalassaemia carrier status and
asked them whether they had been told about such testing.
Of the 146 women asked, 113 said they had not been told
about thalassaemia carrier testing (77.4%) and 97 of these
women (85.8%) said that they would have wanted to be
told that they would be tested (Table 2).
The themes that emerged from the grounded theory
analysis include perceived information needs, the rela-
tionship between information
tion preferences, barriers to obtaining information and
attitudes towards consent for antenatal thalassaemia carrier
testing.
andanxiety, informa-
Perceived pre-test information needs for antenatal
thalassaemia carrier testing
Some women said that they wanted to be informed about
conditions for which they had been tested so that they knew
what their baby was not at risk of having.
Huma (non-carrier, completed questionnaire in Urdu – 19
years old primigravida, six months in the UK): ‘I was worried
about why they took the blood. I should have been told what
for. It’s obvious that they’re taking blood for seeing if there
are any disorders, but we don’t know what for.’
Women particularly wanted pre-test information if they
had relatives with thalassaemia or were aware of their
chances of being a thalassaemia carrier through the media,
Table1 Participants’ demographic details
Carriers
(n¼19)
Possible
carriers
(n¼17)
Non-
carriers
(n¼110)
Age (years)
Median
Range
24
16–43
30
19–38
23
17–37
Gestation (weeks)
Median
Range
20
9–33
19
12–32
22
17–34
Parity (number of women)
Primiparous
Multiparous
12
7
4
13
85
25
Women’s ethnic origin
(number of women)
Pakistani
Indigenous White
African-Caribbean
19
—
—
10
5
2
110
—
—
Women’s self-ratings for
quality of spoken English
(number of women)
Very fluent
Fluent
Okay
Poor
No English
7
—
3
—
9
9
1
1
3
3
47
13
15
9
26
Table2 Whether women were told and whether they wanted to be told about thalassaemia carrier testing
Groups
Carriers (n¼19)Possible carriers (n¼17)Non-carriers (n¼110)Total (n¼146)
Told about thalassaemia carrier testing?
Yes
No
Not applicable?
Do not know/cannot remember
1422
87
—
1
27
113 13
5
—
13
—
—
5
1
Wanted to be told about thalassaemia
carrier testing?
Yes
No
Not applicable??
?Women who initiated the conversation about thalassaemia carrier testing, because they were aware of their carrier status.
??Women were not asked this question if they had said that they were told about thalassaemia carrier testing, or if they said that they did not know or could not remember whether they had
been told
12
2
5
9
4
4
76
11
23
97
17
32
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so that they could discount the possibility of their baby
having thalassaemia.
A number of women also felt that they had an ethical
right to know that they were being tested.
Jackie (possible carrier – 26 years old, multigravida, White
British): ‘Ethically, is it right to test people for something that
they’re not aware that they’re being tested for? y if it is
being tested for, then surely women should have the right to
know about it.’
Relationship between pre-test information and
anxiety
Some women mentioned that provision of pre-test informa-
tion could result in anxiety about conditions that the baby
could have. A possible carrier said that she was not
interested in pre-test information because ‘you start think-
ing too much’. On the other hand, a number of women who
also acknowledged that such information could increase
anxiety said that they would have wanted pre-test informa-
tion. For example, Fozia suggested that women worry about
their baby’s wellbeing regardless of what they are told about
tests. So providing pre-test information does not make
them worry more, it just gives them something else to
think about. She also believed that such worry was not
harmful.
Fozia (non-carrier, interviewed in English – 20 years old,
primigravida, born in the UK): ‘y I mean it’s not as if we’re
not worried anyway. So something like that [pre-test
information], I don’t think it would have mattered to me
even if I was worried about it because I do worry, worry all
through my pregnancy.’
Many women suggested that pre-test information should
be provided regardless of whether it causes anxiety. Instead
of being perceived as harmful for the mother, anxiety was
perceived as an inevitable, but natural, part of being
pregnant. Another woman suggested:
Jackie (possible carrier – 26 years old, multigravida, White
British): ‘yyou need to be told all the information y it does
cause probably more distress ybut that’s just something
you’ve got to go through. It’s part of being pregnant.’
Pre-test information was also seen as a means of reducing
anxiety on receipt of a positive test result. Most of the
carriers and some of the possible carriers said that informa-
tion about being tested would have forewarned them that
they could receive a positive test result and may have
prepared them so that they were not as ‘shocked’ by the
result as they were when they were unaware of being
tested:
Naheed (b-thalassaemia carrier, interviewed in Mirpuri –
21 years old, primigravida, 10 months in the UK): ‘If people
know about the test and what a thalassaemia carrier is then
they wouldn’t be so worried when they find out that they are
a carrier.’
Women’s positive thalassaemia carrier results
accompanied by an unexpected urgent request for their
husband to have a blood test. This may have increased
women’s perceptions of the severity of their positive test
results:
were
Nageena (b-thalassaemia carrier, interviewed in English –
24 years old, primigravida, born in the UK): ‘you must
immediately come to the hospital and get yourself tested and
get your husband testedy And it’s really frightening is that. I
was upset and I was frightened.’
Women felt that pre-test information alerting them
about the implications of a positive test result would have
resulted in comparatively less anxiety than knowing
nothing about the test or the possibility of receiving such
results.
Type,amount and timing of informationpreferred
On the questionnaire for non-carriers, the majority of
women indicated that they wanted pre-test information to
include details about the condition (81%), the test proce-
dure (80%), when the results would be available (80%), the
meaning of positive and negative results (73%), and likely
action following a positive result (77%). However, during
the interviews many women suggested that they did not
want pre-test information to include ‘too much detail’. The
different groups of women interviewed showed a preference
for different amounts and type of information at different
stages in pregnancy.
During the interviews, the non-carriers were less likely to
say that they wanted detailed pre-test information. For
example, some of them did not want any information,
saying that they were happy knowing that ‘everything was
fine’, while some said that pre-test information should
include details about the condition for which they were
being tested, why they were being tested and the possible
outcomes of testing.
Carriers and possible carriers wanted more detailed pre-
test information than the non-carriers. They suggested that
pre-test information should include details about receiving
positive results via a thalassaemia counsellor or letter, and
the implications of being a thalassaemia carrier. Many of
these women clarified that they did not want too much pre-
test information, because there was already a lot of
information being exchanged during the booking session:
Fiona (possible carrier – 32 years old, primigravida, White
British): ‘ythe midwife is getting a lot of information from
you and vice versa so you don’t want to be bombarded with
too much informationy’
Some of the women did not expect midwives to go into
great detail for any specific antenatal test because of time
constraints on the midwife:
Jackie (possible carrier – 26 years old, multigravida, White
British): ‘I’m not suggesting that they have a discussion with
every single woman who goes through antenatal testing.
Obviously that takes up a lot of the time of the midwives, and
obviously their time is precious, they have a lot of women in
their care that they have to speak to.’
Some of the possible carriers highlighted that the issue of
thalassaemia is not salient to them at the time of testing, so
they are unlikely to be interested in detailed pre-test
information. They also suggested that pre-test information
could be best provided in the form of a leaflet which they
could read immediately or refer to on receipt of a positive
result. Women from all three groups suggested that the most
appropriate time for detailed information and explanations
was on the receipt of a positive test result. They suggested
that information provided at this stage should include details
about the condition, why the woman is a thalassaemia
carrier, implications of being a carrier, why the father has to
be tested, implications for the baby and implications for
other children.
Perceptions of ‘information’ and ‘consent’ 73
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Barriers to acquiring information
Many women said that they did not know enough about the
condition and/or test to ask health professionals any
questions:
Fatima (non-carrier, interviewed in English – 25 years old,
primigravida, born in the UK): ‘I just go to see her [midwife],
but I don’t ask many questionsy but it’s my first time yeah,
I don’t really know the questions.’
In addition, some women said that they did not ask for
information because they believed that health professionals
would automatically provide it for important tests.
Fozia (non-carrier, interviewed in English – 20 years old,
primigravida, born in the UK): ‘I didn’t [ask about tests]
because I mean I thought well, you know, it’s my midwife
and if it was something important, maybe she would tell me
herself and that I wouldn’t need to ask.’
Most women said that they did not know which tests
were important and so left the decision of providing
information about important tests to health professionals.
Some of the women who were unable to speak English
compromised their information requirements. They per-
ceived not being able to speak in English as an obstacle
placed by them in the process of communication. So, instead
of expressing a desire for information, they accepted that
they could not receive or obtain information:
Zora (possible carrier, interviewed in Mirpuri – 38 years
old, multigravida, 10 years in the UK): ‘yI don’t speak or
understand English properly. This is my problem [her
emphasis]. I did ask the midwife why she was taking five
bottles of blood. She did say something, but I don’t know
what because I didn’t understand.’
However, given a choice, non-English-speaking women
would have preferred some pre-test information:
Lubna (possible carrier, interviewed in Mirpuri – 19 years
old, multigravida, four years in the UK): ‘Yes [would want
information in own language]y it’s obvious that one can
then do everything for oneself. If you understand, then you
could ask questionsy’
The following quote shows how difficult it was for a non-
English-speaking woman to obtain information, even
though she had a strong need and desire for it:
Abida (possible carrier, interviewed in Mirpuri – 30 years
old, multigravida, 10 years in the UK): ‘y I’m having twins
yI don’t speak English. My husband usually goes with me,
but he never explains anything to me properly. They explain
everything to him, but he only tells me a little of what he’s
told. I want things to be explained to me y I really want to
know what’s going ony I want to know more. I want to ask
more.’
If it is so difficult to ask for information when it is desired
and needed, then it is likely to be more difficult when
women do not know what tests are being performed or
what questions to ask. The above quote also shows that
health professionals should not assume that an individual
will receive sufficient information, satisfactorily, via a
relative who interprets for them, or that individuals would
be able to ask for information they require via such an
interpreter:
Jameela (b-thalassaemia carrier, interviewed in Urdu – 22
years old, multigravida, two years in the UK): ‘So when the
midwife came, I said to my sister-in-law that ‘‘she is taking
my blood so ask her about thalassaemia’’. She [sister-in-law]
said that ‘‘if there is [thalassaemia] then she will tell you
herself’’, so I said ‘‘okay’’.’
Jameela did not know that she had been tested until she
received her positive test results.
Another reason women may not ask for information is
because they believe that there is no need since their
antenatal notes, which they keep and take to antenatal
clinics, will be able to tell them what tests they have had.
For example, a non-carrier interviewed immediately after
receiving her routine antenatal test results looked through
her notes to find out whether she had been tested for
thalassaemia carrier status. Her notes showed that she had
had the ‘haemoglobinopathy screen’ and the result was
‘sickle cell screen – negative’ and ‘no abnormal haemoglo-
bins’, but thalassaemia was not specifically mentioned. So
while some women believed their notes to be a source of
information, they were unable to extract information about
thalassaemia carrier testing because they did not understand
what was written. These findings suggest the need for
clearer reporting of negative test results.
Consent for antenatal thalassaemia carrier testing
In response to a question in the questionnaire, 129/146
(88.4%) of the women said that they were not asked for
their consent for thalassaemia carrier testing (Table 3).
Attitude towards consent for thalassaemia carrier
testing
Most women who knew that they had been tested believed
that thalassaemia carrier testing was routine and that they
had no choice about testing. Some women believed that if
they had the right to choose, their midwife would have
asked them. However, many women acknowledged that
they had insufficient knowledge about various conditions or
tests to make decisions about which ones they should be
tested for:
Yasmin (b-thalassaemia carrier, interviewed in English –
22 years old, primigravida, born in the UK): ‘They [women]
don’t know what thalassaemia is and whether they should be
having a test for it, but the doctors doy’
Table3 Whether women were asked for their consent for thalassaemia carrier testing
Groups
Asked for consent to thalassaemia carrier testing? Carriers (n¼19) Possible carriers (n¼17) Non-carriers (n¼110) Total (n¼146)
Yes—
No 14
Do not know/cannot remember5
37 10
12914
—
101
27
74Ahmed et al.
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Many women viewed doctors and midwives as trained
experts, who were competent in caring for mothers and
babies. They suggested that because health professionals
‘know best’ and because women had comparatively little or
no knowledge of the condition, the health professionals,
rather than the pregnant woman, should decide which
conditions she should be tested for:
Haleema (b-thalassaemia carrier, interviewed in English –
19 years old, primigravida, born in the UK): ‘No I don’t think
you can pick and choose [which tests you have]. What would
women know about, you know, the doctor knows morey’
Another reason women allowed health professionals to
carry out various tests without understanding much about
them was because they trusted health professionals:
Sadia (non-carrier, interviewed in Punjabi – 22 years old,
primigravida, seven months in the UK): ‘yobviously what-
ever tests they do, they would think that it is better for usy If
they think it is better to do these tests, then it’s okay, let
them.’
Women perceived doctors and midwives in a caring role,
as well as in a position of power and authority, and therefore
placed themselves in the health professionals’ hands,
entrusting them to do whatever they thought was best:
Interviewer: ‘So do you think that the way in which they
tested you [without any information] was right or not?’
Jameela (b-thalassaemia carrier, interviewed in Urdu – 22
years old, multigravida, two years in the UK): ‘I think that
they have done very well to tell me (that I am a carrier). At
that time I thought ‘‘how good these people are, that they
care about us so much [kithna khayal rakhte hein], because
it’s obvious they did it for usy’’’
Three women were unhappy about being tested for
thalassaemia carrier status without having given prior
consent – all were articulate and professional women and
knew that they had been tested before meeting the
researcher. Only one non-carrier was genuinely concerned
about consenting to antenatal thalassaemia carrier testing,
and even this was because she worked in the legal field and
was aware of her right to consent to testing. Furthermore,
only two of these women said that they would have refused
thalassaemia carrier testing. In one of these cases, the
woman was already aware that she was a carrier and that
her husband was not. She was angered about being re-
tested because she had specifically agreed with her midwife
not to be tested again and believed re-testing to be a waste of
her own and health professionals’ time and resources. In the
other case, the possible carrier (in her third pregnancy)
revealed that she only became concerned about not having
given prior consent to testing when she realized that the test
had been done as a matter of routine, and not at her
midwife’s request.
Shazana (Possible carrier, interviewed in English – 35 years
old, multigravida, born in the UK): ‘ywhen I went back to
her [midwife], she was the one that said to me ‘‘oh, I didn’t
know you’d been tested for it.’’ ythat made me feel even
worse’.’
Importance oftesting versus consent
Many women also suggested that it was better for health
professionals to test for thalassaemia carrier status without
obtaining their consent prior to testing, than not to test
at all:
Zakia (b-thalassaemia carrier, interviewed in English – 23
years old, primigravida, born in the UK): ‘I mean if they
know it’s quite common and they test you anyway [without
asking], at least you know you’ve got it or you’re carrying it. I
wouldn’t have known just to go and get myself tested for it.
And if they’ve done it, fair enough, at least I know, it’s better
than not knowingy’
Interviewer: ‘Do you think they should ask?’
Zakia: ‘I don’t know. It doesn’t bother me though. It’s
something that I need to know.’
Many of the women felt that it was important to know
whether there was anything wrong with them or their baby.
Therefore, women approved of health professionals testing
them for whatever conditions they considered to be
important, without obtaining prior consent. One carrier
thought that antenatal testing for thalassaemia was so
important that she suggested that women should not be
offered the choice of whether they wanted thalassaemia
carrier testing because they may refuse to have it through
lack of understanding about the importance of the test.
DISCUSSION
In view of the NHS Plan for commitment to a national
antenatal and neonatal screening programme for thalassae-
mia and sickle cell in 2004, this is a timely study which
aimed to explore the issues of informed consent and
perceived pre-test information needs within a pregnant
population. The strengths of this study are that (a) it is the
first to explore attitudes towards informed consent for
antenatal thalassaemia carrier testing, (b) the sample
includes women with a variety of experiences of antenatal
carrier testing and (c) it includes data from Pakistani women
regardless of literacy or ability to communicate in English.
The present study, like a number of others,22–25has shown
that there is a gap between the information women want
about tests during pregnancy and the information provided
by health professionals. Although the data in the present
study were collected from women an average of two months
after the event, and therefore could reflect faulty recall, they
accord with observational studies26–28and support the
pattern of results reported elsewhere,29including a Health
Technology Assessment review on ‘Psychosocial aspects of
genetic screening of pregnant women and newborns’,
which very clearly shows that lack of understanding within
this context is widespread.30
The findings also show that non-English-speaking women
refrained from requesting information in a different lan-
guage. There may be a number of reasons for this. One
reason is that assertiveness in a medical context is culturally
perceived as inappropriate, and women did not want to be
seen as lacking confidence in their midwife/doctor or as
questioning their authority by asking for information.
Another reason for not asking questions may be the fear
of being labelled as troublemakers.31,25Women may feel
that they are dependent on health professionals for
antenatal care, childbirth and postnatal care, and are likely
to want to avoid being labelled a troublemaker by refraining
from asking for information in a different language. In
addition, women may not want to be seen as ‘special needs
cases’, or as creating extra work for their midwife. During
the interviews the researcher observed that most women
were reluctant to say anything that could be interpreted as
critical of midwives or antenatal services. The researcher felt
that women genuinely appreciated the antenatal services
available ‘to the extent that they did not feel that they had
Perceptions of ‘information’ and ‘consent’75
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................
Authors’ affiliations
Shenaz Ahmed, Research Fellow, Academic Unit of Psychiatry &
Behavioural Sciences, University of Leeds, Leeds LS2 9LT, UK
Josephine Green, Professor of Psychosocial Reproductive Health,
Mother & Infant Research Unit, University of York, York Y10 5DD, UK
Jenny Hewison, Professor of the Psychology of Health Care,
Academic Unit of Psychiatry & Behavioural Sciences, University of
Leeds, Leeds LS2 9LT, UK
the right to hold, let alone state, their individual prefer-
ences’.31Like others,19the present study found that even if
women know their information needs, they did not always
know how to communicate them. Overall, women dis-
played compliant behaviour, where desire for information
came second to building up and/or maintaining a good
relationship with the midwives. This should not be inter-
preted as women not wanting information, just that they
did not feel they could ask for it.
The main aim of the study was to explore pregnant
women’s views about informed consent. The findings
showed that the concept had little meaning for these
women. Instead, women talked about ‘information’ and
‘consent’ as separate issues, where most of them wanted to
be informed about testing but did not necessarily want to
consent, partly because of their views on who should make
such decisions. That is, many of the women said that they
trusted doctors and midwives because of their perceived
expertise and competence in caring for mothers and babies,
and believed that since health professionals ‘know best’
then they, rather than pregnant women, should decide
which antenatal tests should be carried out.
While most women wanted to know that they were being
tested for thalassaemia carrier status, they did not expect
their consent to be obtained prior to testing. This may be
because women were not aware of a right to choose to have
or not have thalassaemia carrier testing. Like other routine
antenatal tests, they did not expect to be involved in
decision-making about whether or not they should be
tested. Furthermore, the findings show that most women
did not want to make decisions about antenatal thalassae-
mia carrier testing: they preferred to leave such decisions to
the health professionals instead. This could be because they
had poor knowledge about the conditions being tested for,
and also because of the trust they had in their health
professional as the decision-maker.32Others suggest that
women believe in the knowledge and competence of health
professionals, placing them in a position of authority and
allowing them to make decisions about health care.19
However, some of the women in the present study stated
that they would have preferred to be given choices about
antenatal tests and would have wanted to make decisions
themselves. Therefore, health professionals could provide
basic information about the conditions for which antenatal
testing is being offered and determine the extent to which
women want to be involved in decisions about which tests
to have.
The majority of women showed that they were not
concerned about giving consent for antenatal thalassaemia
carrier testing. However, it is not clear how they would feel
about consent for antenatal testing for other disorders, such
as cystic fibrosis, Down’s syndrome or late-onset conditions.
Overall, there are a number of implications for service
provision. Schemes such as ‘Informed Consent for Antenatal
and Neonatal Screening’ (commissioned by the National
Screening Committee) and ‘PEGASUS’ (a training network
commissioned by the NHS Sickle Cell and Thalassaemia
Screening Program) to develop training for health profes-
sionals to facilitate the implementation of the Program, with
a focus on training for frontline health professionals (such as
midwives and GPs), are currently in the pipeline. Our
findings show that such training should include information
on laboratory investigations for thalassaemia carrier status,
showing how the first stage of this investigation involves a
routine MCH measure that is performed for all women. This
would enable frontline health professionals to understand
how and why women could be tested for thalassaemia
carrier status even if they do not request such testing.
Furthermore, there is a need for guidelines that allow
laboratories to identify women who have not consented to
testing following the initial routine MCH screening test, and
then specify how to obtain consent so that women who
have not already been approached can opt out of further
testing at this stage if they wish to do so.
In relation to information provision and people’s needs for
varying amounts of information, one way forward could be
for health professionals to assess pre-test information needs
on an individual basis and provide information accordingly.
However, there are problems with this because of the time
constraints on health professionals. An alternative is to
provide information in written form (i.e. leaflets in different
languages)33
so that women can extract information
according to their own needs.
Leaflets about being a thalassaemia carrier and how
thalassaemia is inherited are currently available, which have
been developed by the UK Thalassaemia Society34and by
individual health authorities. However, the language used
in both the English and particularly the Urdu version is
complicated and sometimes misleading. For example, in
their review of genetic information available in Urdu, Shaw
and Ahmed33found inaccuracies in leaflets differentiating
carriers from affected individuals. There is a need for a
leaflet specifically designed for antenatal thalassaemia
carrier testing (initially in English and then in other
languages). Such a leaflet should be evaluated for its ease
of accessibility, its ability to meet women’s perceived pre-test
information needs and its ability to serve as a reference
point for women receiving positive results. Perhaps separate
leafletsshouldbe developed
a-thalassaemia carriers in order to reduce the confusion
that could arise from presenting the two in the same leaflet.
Women with information needs exceeding those provided
in the leaflet could be referred to specialists, such as
thalassaemia counsellors, instead of expecting midwives to
provide detailed information. Of course, leaflets are only
useful if women can read them. For women unable to read,
yet another alternative would be to provide information in
the form of audio or videotapes.
for
b-thalassaemiaand
CONCLUSION
The analysis has allowed the deconstruction of the concept
of informed consent for these women. The findings show
that while information was important to women, consent-
ing was not. Overall, women discussed information and
consent as two separate issues, thus challenging many of the
assumptions around the term informed consent. They
wanted pre-test information because they just wanted to
know, not to use it to make decisions about testing.
ACKNOWLEDGEMENTS
This project was funded by the University of Leeds under
their Postgraduate Research Scholarship scheme.
76 Ahmed et al.
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