Social Science a Medicine 54 (2002) 1333–1344

Attitudes to bioethical issues: a case study of a screening

project
U. Gustafsson Stolta, P.-E. Lissb, T. Svenssonc, J. Ludvigssona,*
a
Department of Health and Environment, Division of Paediatrics, Faculty of Health Sciences, University of Linko.ping,
S-581 85 Linko.ping, Sweden
b
Department of Health and Society, Linko.ping University, S-581 83 Linko.ping, Sweden
c
Department of Behavioural Sciences, Linko.ping University, S-581 83 Linko.ping, Sweden

Abstract

Commonly expressed in theoretical discussions about ethical problems in the context of epidemiology and screening
is the need for more data. A study was carried out involving 21 explorative interviews with participant and
nonparticipant mothers in a neonatal research screening project in progress in Sweden, ABIS (All Babies in Southeast
Sweden). The respondents were asked, by way of open-ended questions, to give their opinions about certain ethical
issues: informed consent; reasons for joining/declining; surrogate decision; the collection, analysis and storage of
written and ‘‘live’’ material (biobanks); intervention etc.
The ethical implications mentioned in the literature mostly concern the risk of creating distress and anxiety (anxiety
and possible stigmatisation in respect of positive or false-positive results, worry about material collected and stored,
distress caused by blood sampling procedures, etc.). Our results do not support the idea that the risks are substantial.
The respondents rather indicate an attitude of benevolenceFthey are positive both to the current research on children,
to the material they contribute (both written material and ‘‘biomaterial’’), to possible results and intervention plans. On
the other hand the participants expressed concern about the storage of material and the right to be informed of any
screening/project results. Further studies in this field are needed and would be of help in theoretical discussion, the work
of ethical committees and the designing of, for example, screening and research projects. r 2002 Elsevier Science Ltd.
All rights reserved.

Keywords: Ethics; Screening; Neonatal; Informed consent; Diabetes; ABIS; Sweden

Introduction Faden, Chwalow, a Horn, 1983; Faden et al., 1985;

Neonatal screening is performed routinely in many
countries, including Sweden, in order to prevent some
severe diseases. In Sweden routine samples are for
example taken for phenylketonuria, hypothyroidism and
galactosemia. There are some studies discussing relevant
ethical issues such as for example the complexity of
informed consent, the relationship between information
and understanding/knowledge, the issues of positive
results (Faden, Holtzman, a Chwalow 1982; Holtzman,
*Corresponding author. Tel.: +46-1322-1333; fax: +46-
1314-8265.
E-mail address: johnny.ludvigsson@lio.se (J. Ludvigsson).
Bradley, Parsons, a Clarke, 1993; Parsons, Bradley, a
Clarke, 1996; Fenton-May et al., 1994, etc.). Similar and
other questions rise when screening are done for
research purposes.
The importance of cooperation and understanding
between (and of) the public/participants and medical
research is crucial in the area of screening projects,
perhaps particularly in the case of major research
screening of neonatals and their families over several
years. During the last ten years two major projects have
commenced in Europe, the ALSPAC (the Avon Long-
itudinal Study of Pregnancy and Childhood) and ABIS
(All Babies in Southeast Sweden). Even though they
differ on some points, for example in questions of

0277-9536/02/$ - see front matter r 2002 Elsevier Science Ltd. All rights reserved.
PII: S 0 2 7 7 - 9 5 3 6 ( 0 1 ) 0 0 0 9 9 - 5
1334 U. Gustafsson Stolt et al. / Social Science a Medicine 54 (2002) 1333–1344
anonymity and therapeutic measures, the two projects
are both designed as research screening projects of
neonatals and their parents, who have been and will be
closely followed over several years. The similarities are
many, not only in number of participants, methods,
general aims, etc., but also in the way they focus on the
many ethical issues that are involved in research on
children (Golding, 1989; Golding a The ALSPAC
study team, 1996; Mumford, 1999a, b). Even though
there is a common acceptance of the validity of the basic
general ethical principles advocated by Beauchamp and
Childress (1994), there are still questions that need to be
answered as to, for example, how and when these
principles are to be used in practice. The ethical issues
concerning screening can roughly be described as
consisting of three kinds in this context: how can we
do good without causing (or at least how to minimise)
harm, how can we do good without infringing autonomy
and integrity, and how can we do good without being
unfair? In research the result is often a question of
balancing: issues of balancing the ethical concerns in
those cases where the goods are being judged as
sufficiently great to justify an intervention, for example
the ABIS project.
Screening normally involves certain risks for the
participants involved. There are risks for false positive
results and the creation of distress and anxiety (Schulte,
1985; Schulte a Singal, 1996; Holtzman, 1988;
Hermer!en, 1999). There are risks of bypassing informed
consent and stigmatisation (Fost, 1999). An important
issue is also the question of results and information
about them (Brody, 1997; Childress, 1997; Mumford,
1999a, b; Walters, 1997). And there are risks concerning
storage of obtained material. However, these are only a
few examples. The point here is that the discussion
about how large these risks are and how to avoid them is
built on empirical premises which are to a large extent
insufficiently studied (Hermer!en, 1999; Riis, 1999).
Significant empirical knowledge is to a large extent
lacking in the field of descriptive ethics (Holm, 1996).
Concerning research screening there is, for example,
limited knowledge concerning both the attitudes of the
participants of the project and why people choose to
participate or not. Regarding the latter, is it for ethical
reasons or some other reasons? We also need more
knowledge to what extent the decision to participate or
not is sufficiently voluntary and well-informed, or to
what extent a particular screening study causes distress
and anxiety; it is a matter of feelings about the actual
participation, about the samples and information they
provide, but also about possible results and notification
thereof. Concerning research screening of neonatals
there are some studies exploring these issues (see, for
example, Smith, Williams, Sibert, a Harper (1990) and
Bradley et al. (1993) concerning screening for Duchenne
muscular dystrophy). But still some of these issues and
others related to them need to be further described and
investigated into. Concerning, for example, the impor-
tant issue of informed consent, there is limited data
concerning actual consent practices, with the result that
guidelines, codes, etc. depend heavily on theory (Beau-
champ, 1997).
In short, we need to know more of the perspectives of
the individuals involved and what they consider to be
‘‘ethical problems’’. We also need knowledge of how
these participants in projects perceive the ‘‘study
design’’, i.e. in this case safeguards against ethical
possible dilemmas, since it already been decided for
them. This particular question is on the agenda of the
ALSPAC’s own ethical committe for a future study, a
study that will further help to increase our knowledge in
this area (Mumford, 1999b).
ABISFAll Babies in Southeast Sweden
The main purpose of the ABIS project is to predict
Type 1 diabetes (juvenile diabetes) in the general child
population and to prospectively study the importance of
environmental factors for the development of Type 1
diabetes, together with some other so-called autoim-
mune diseases such as celiac disease and inflammatory
bowel disease (IBD). Between October 1997 and
October 1999 all 21,700 mothers-to-be in the southeast
of Sweden received information about the project and, if
they consented, their participation began when the child
was born. The information, both oral and written, was
given at least two times during their pregnancy. Our
second study of staff interviews has shown that the
information was given at 12 weeks of pregnancy resp.
23–30 weeks. A video and a poster (later also a
broschure) was also produced for the purpose of
information. Approximately 16,000 children and their
parents are participating in the project, which is 75% of
those 21,700 newborns and their families asked to
participate. Several methods are used in order to achieve
the projects main aims; samples from the child and
parents (blood, hair, breastmilk, etc.), questionnaires to
be filled in certain time intervals (first at maternity ward,
then at intervals of 1, 2.5–3 and 5–6 years), a diary where
the parents during the child’s first year note down food
consumption, infections, medical treatments, vaccina-
tions, etc. Another purpose is to try to identify high-risk
individuals from the material collected, the aim is to
develop methods for identifying those children that can
be diagnosed as having a higher risk of developing child
diabetes. If the methods prove successful one further aim
is to make it possible to offer those children that have
been identified as having higher risk and their families
any available preventive measures. The ‘‘counter-
measure’’ here mostly discussed is the B-vitamin
Nicotinamide.
 U. Gustafsson Stolt et al. / Social Science a Medicine 54 (2002) 1333–1344 1335

The objective of this paper is to explore and describe Table 1

ethically relevant opinions and attitudes among a Project participant characteristics
sample of participants and nonparticipants in respect Characteristics Number
of the ABIS project.
(a) Category 1Fparticipants in the ABIS project (n ¼ 15)
Age
Methodology o25 2
26–34 7
>35 6
Participants
Marital status
Single 0
The sample for our qualitative study consisted of 21 Living in couple 15
women who had just become mothers, 15 of whom Education
consented to participate in the project, which reflects the oO-level 7
proportion of mothers who accepted or declined to Higher educationFuniversity 1–3 7
participate in the main study. We did not choose a >University 3.5 years 1
representative material, but made a strategic selection Parity
concerning both categories of respondents. The vari- First child 6
ables age, education, marital status and parity were Second or more 9
(b) Category 2Fnonparticipants in the ABIS project (n ¼ 6)
utilised to reflect a broad range of different types of
Age
mothers with different backgrounds (Trost, 1997). An o25 1
attempt was made to incorporate mothers with a foreign 26–34 4
background and single mothers, but this proved >35 1
unsuccessful. It was impossible to use a similar selection Marital status
strategy where the nonparticipant mothers were con- Single 0
cerned due to the fact that they were (and are), only Living in couple 6
listed as nonparticipant in ABIS at the child welfare Education
centre. The sample of six mothers was selected with the oO-level 4
aid of staff from one such centre. However, the category Higher educationFuniversity 1–3 1
>University 3.5 years 1
two sample shows a satisfactory variation when
Parity
compared to the first category of respondents. Back- First child 3
ground information on all study participants cited in Second or more 3
this paper is contained in Table 1.
After a preliminary analysis of the 21 interviews we
found a saturation regarding information and opinions
among the respondents. We decided that further inter- was taken to assure the participant mothers that the
views would give no additional information. present study did not in any way influence their
The average age of the total number of respondents participation in the larger ABIS project.
was 32, ranging from 21 to 44. Since no single mother The data collection, by way of semi-structured inter-
agreed to participate in this particular project, 100% are views, was accomplished during the autumn of 1999. A
either living together or married. Half of the respondents total of 21 interviews were completed with participant
have basic A-level (Swedish gymnasium) as highest and nonparticipant mothers. To qualify for the study the
degree of education, the other half university studies mothers had to have given birth not later that six
ranging from 1 to 5 years. Fourteen of the participant months before interviewed, to ensure that the shortest
mothers, including the six nonparticipant have in ABIS time possible had passed since the initial first informa-
their first personal contact with a large medical project, tion was given to them about the ABIS project (resulting
the remaining one mother had her previous child also in that all interviews where completed between 3 and 6
participating in the ABIS project. Nine of the 21 months after delivery). The interviews were all con-
respondents are mothers for the first time, the youngest ducted individually and in the respondent’s own home.
21, the eldest 37 years of age. The length of the interviews varied from one to three
hours. All interviews were tape-recorded with the
Procedure (data collection and analysis) consent of the participants.
The interviews started with open-ended questions
A letter of information was sent to the mothers in (Patton, 1980; Kvale, 1997) about ABIS (general
which they were asked if they would consent to questions, for example what information they had
participate. After initial contact interviews were con- received). The content of the interviews was decided
ducted at a time and place of their choice. Special notice beforehand on the basis of literature and our aims of the
1336 U. Gustafsson Stolt et al. / Social Science a Medicine 54 (2002) 1333–1344
study (for example, Veatch, 1989; Beauchamp a
Childress, 1994; Childress, 1997). Questions concerning
ethical issues were avoided in order to get personal
spontaneous comments which could then be comple-
mented by follow-up questions. A series of topics/
subject areas were then successively introduced, provid-
ing a common structure for all the interviews. These
included issues concerning the context of information
and decision, reasons for joining/declining, surrogate
decision, the material and information collected for
ABIS, possible results of the project and issues related to
questions of prevention/intervention.
The interviews were transcribed in extenso. A primary
analysis of the transcripts was then made, providing a
coding according to categories based upon the interview
themes. Several reviews of the transcripts were made
during this stage of the analysis to ensure that all relevant
data were subsequently accounted for. In a second round
of analysis the initial theme categorisation was refined and
the material was structured according a common
recognised theoretical bioethical framework (Beauchamp
a Childress, 1994). An attempt has been made to
facilitate an ethical discussion and a more theoretical
analysis. We would also here like to comment on a
theoretical categorisation made in this paper refering to
the notions of concern respectively distress/anxiety. By the
term ‘‘concern’’ we refer to a cognitive activity. A person
who is concerned about an ethical issue is pondering on
the issue, which may or may not lead to distress. By the
term ‘‘distress’’ and ‘‘anxiety’’, on the other hand, we refer
to different degrees of emotional states.
An important point to make is that this is a
qualitative study and although numerical data are
presented, we are aware that the data should be looked
upon with caution, the fact being that the sample has
been chosen not to be statistically representative or
randomised (in order to reflect widest range possible).
On the basis of the results of this explorative study a
larger-scale quantitative study, involving questionnaires
presented to a statistically representative sample of 400
participant mothers, is under way. The findings from
that study will be presented in a later paper.
Results
General attitudes towards screening research on children
Both categories of respondent were asked how they
perceived this kind of research on children, what
opinions they had about it and, if they were participants,
what they thought regarding their participation in the
project. All 21 respondents displayed a positive attitude:
For myself, I am very interested in health. If you can
help this kind of research, and it’s successfulFthen it
is important and good. It is also very good for my
own sake, for example it will help me to see the
connection between food and illness when my own
child gets ill (Participating mother 5 (ID), 29 (age):
1 (Parity, 1st child).
All of the participant mothers thought it was
important that they and their family participated in
the study. They were asked in what way they felt it to be
important. To illustrate the predominant view:
I feel important, research cannot progress without
people willing to be a part of it. It feels important
that the information I give is completely correct, even
though it is difficult at times. But I thought in terms
of I will do my best (Pmo 8, 29:2).
As is also shown by the reasons the participating
mothers have given (Table 3), there is an explicit wish to
contribute to research:
The advantage of participating in ABIS is that you
can contribute to research, to help find out why
certain diseases occur here, andyy (Pmo 3, 33:3).
The same positive attitude is visible in the case of the
nonparticipating mothers. Even though they have
different reasons for not accepting the invitation, their
overall attitude to screening research on children is
positive:
We both have a disease and we are both fighting it, so
you are grateful for the things done earlier in research
and thenyy.you want to give what you can, help in
the way you can. I suppose the advantage is that you
feel useful, even though your own child should
become ill you know you have done something,
butyyyyy. (Npmo (nonpartcipating mother)1,
31:2).
Information
The mothers-to-be were informed about the ABIS
project at approx. 12 and 23–30 weeks of pregnancy.
The decision was to be made (and was asked for) at the
delivery ward where also the first samples of blood were
taken; cord blood from the infant and blood samples of
initially the mother (later also the father was asked to
contribute). As shown in Table 2, 9 of the 15 participant
mothers say they recall receiving this initial oral
information (at the maternity ward) about the project.
Among the nonparticipating mothers 5 out of 6 recalled
having been informed by their midwife during their
pregnancy. As shown in Table 2 the information were
structured in different ‘‘types’’: oral, written and
audiovisual (a video was produced with the intention
of playing it for the mothers, a poster to inform
 U. Gustafsson Stolt et al. / Social Science a Medicine 54 (2002) 1333–1344
Table 2
Factors related to recollection of information
Statements of recalled information received during Number
pregnancy (decision asked at delivery ward)
(a) Category 1Fparticipants in the ABIS project (n ¼ 15)
MVCFMaternity welfare
Oral 9
Written 7
Audiovisual (video-poster) 0–4
Information sufficient for the decision made
Yes 15
No 0 with the participating mothers was the way they
(b) Category 2Fnonparticipants in the ABIS project (n ¼ 6)
MVC–Maternity welfare
Oral 5
Written 5
Audiovisual (video-poster) 0–0
Information sufficient (about the project itself)
Yes 5
No 1 If you, through this, can help in any wayysoyit can
Never asked to make a decision 3 give something, then you are only too happy to help
Would have participated if asked 1 and contribute. I don’t consider there’s to much work
mothers-to-be in the waiting room or another public
area. Together with the oral information the women
were presented with more detailed written information.
According to the recollection of eight of the participant
mothers, no letter of information was received by them
(or read). One possible practical reason for this can be
the amount of information given to the mothers on their
first visit to the maternity welfare, which a number of the
respondents mention. Of those seven participant
mothers four recall receiving additional oral information
at the delivery ward before they made their decision to
participate, three also state that they remember having
received the letter of information later at the maternity
ward. Of the nonparticipating mothers 5 of 6 state their
recollection of having seen/read the letter of informa-
tion. To the category audiovisual information belongs a
video produced by the project management, to be shown
to the mothers-to-be at the different wards. According to
the respondents, none (0) recall having seen it.
The participant mothers were also asked if they recall
the information received sufficient where their decision
was concerned. All respondents claim to have received a
sufficient amount of information for enabling them to
make their decision. They also answered yes to the
question ‘‘Did you understand the information given to
you?’’. Among the nonparticipating mothers five experi-
enced that they, at that time, received sufficient
information about the project itself and this opinion
was expressed also at the time of the interviews, i.e. that
they felt they had sufficient knowledge as to their
participation in ABIS is concerned. As can be seen in
Table 2 the phrase ‘‘for decision made’’ has been
1337
omitted, three nonparticipating mothers say they have
not been asked, i.e. never asked for their decision, either
at the delivery or at the maternity ward. However, only
one of them would have participated, if asked. The one
mother that does state not having received enough
information was asked for her decision at the delivery
ward.
Decision to participate or not
An interesting common theme across the interviews
described the main reason(s) for their participation in
the ABIS project. Instead of predominantly thinking in
terms of what they and their child might gain from their
participating, the most common reason given was a wish
to contribute. As one of the mothers describes it:
involved, it feels more like ‘‘of course you should’’
(participate), if it is to do any good. It is a good thing
and I find no reason not to be a part of it (Pmo 5,
29:1).
Also frequent is the description by the mothers of
their own personal experiences of the diseases focused
upon by the ABIS project, these experiences being a
reason for wanting to participate. They provide exam-
ples of how common these diseases are amongst families
today. Seven of the participating mothers have personal
experience of diabetes, allergy, etc., either themselves or
in their closest family (spouse, parents):
You see, I am afraid that he will develop allergy,
since I have iyand it is not an easy thing when you
get diabetes, because it is a terrible diseaseFmy
mother has it and my grandfather had type 2-
diabetesFand that was, I presume, another reason
for me to join this since we have it in the family. It is
a terrible disease to have, no one thinks so, they think
it’s easy to live with, but it’s not any fun to have. And
a lot was just that, that they’d be doing research
about it, investigating into it. It is terrible that it is
augmenting. And this is what I am very interested in
knowing, what it might be caused by (Pmo 2, 44:2).
While the reasons given by the participant mothers
can be said to have a common ‘‘core theme’’, i.e. help
(help research, help my own child, help other children,
etc.), reasons given by the nonparticipating mothers are
more varied. One reason, the most frequent, is concern
about the blood sampling. Not when themselves were
concerned, but their child. As two of the mothers
illustrates it:
1338 U. Gustafsson Stolt et al. / Social Science a Medicine 54 (2002) 1333–1344
We agreed, my husband and I, we were not going to
take part. And yI was perhaps five years old when
they started to take tests on me because they thought
I had something wrong with my kidneyyandy.I
don’t think you should expose children unnecessar-
yyyto a lot of tests. Even if it’s only one ‘‘jab’’. We
did not want to expose him to that (Npmo 4, 23:1).
No, I would have said no to participate. My son
should not have to go through blood testingsy..I do
not want that and that is why we would have said no
if we were asked (Npmo 5, 35:1).
Table 3
Factors related to reason for decision to participate/decline,
voluntariness , influence
Factors related to decision making
(a) Category 1Fparticipants in the ABIS project
Main reasons stated for decision to participate in the project
A wish to contribute to research
Ilness in family (diabetes, allergy, rheumatism,
astma, celiaci)
A wish to detect if my own child will develop
diabetes, allergy, celiaci, rheumatism etc
Can be of general help to my own child
(development of medicines, discover connection
between food and illness, etc)
A wish to help other children and families
Voluntary decision
Yes
No
Feeling/experience of external influence
Yes
No
(b) Category 2Fnonparticipants in the ABIS project (n ¼ 6)
Main reasons stated for declining to participate
in the project
(Table 2Fone would have participated if asked)
Not wanting to put the child through frequent
testing (blood)
Not wanting the child to be a part of ‘‘experiments’’ 2
A feeling of too much work involved/lack of time
Not wanting to know any results (discovery of
illness)
A feeling of a ‘‘anonymous mass project with
only diagrams and statistics as results’’
Bad experiences of projects/healthcare due to
chronic disease in family
Not enough information received (would most
likely have said yes)
Voluntary decision
Yes
No
Feeling/experience of external influence?
Yes (by spouse)
No
Two of the respondents say that they were concerned
about their child being subjected to ‘‘experimentation’’:
We discussed it, my husband and I, and, wellyin a
way it is good they do research, but we did not want
T to be a guinea-pig or something of the kind. Now
afterwards, one believes it positive to find things out,
but at the same timeyyou might not want to expose
children to a lot of experimentation, that was how it
felt then (Npmo 3, 30:1).
One of the mothers express another reason why she
and the child’s father turned down the invitation to
participate in the study: they did not want to know any
results of their child’s participation:
We said right from the beginning that we did NOT
want to be a part of this because we do not want to
know. It is not the fear of knowingyit is just that,
neither one of us wants to know or find out. Why do
Number
you want to know what will happen? You can find
(n ¼ 15) out if you have cancer genes, and you might need to
do that, but why? If I get it, I get it, I cant protect
14 myself from it. I won’t be happier to know. If you are
6 a part of this kind of research, if you participate, you
want to know the result (Npmo 4, 23:1).
5
Important factors in the context of ethical analysis are
5 the questions of voluntariness and influence. As is
shown in Table 3 all (15) participating mothers concur in
2
their recollection of having made a completely voluntary
decision with no experience of external influence, for
15 example by spouse, parents or nursing staff.
0 However, two of the nonparticipating mothers stated
at the time of the interview that they were partly
0 influenced by their husband in making their earlier
15 decision not to participate in the ABIS project, but when
the decision was made, it was voluntary and not
(n ¼ 5) something which can be said was forced upon them.
The following statement by one of these mothers
4
illustrates this:
When I read about it, I thought ‘This is good, of
course we will participate’, so I had my mind setting
2
1
on us doing it, then. But it was not to be. He thought
it was obvious that they should not. OK, I respected
1 that, so the reason why, well, to be honest a part of me
was comfortable in the decision not to join so I can’t
1 just blame him. But it is with a bad conscience I say
No, but it is not so very easy either..... because I also
1 think.....we are different as personsy (Npmo 1, 31:2).
(n ¼ 6)
6
0 Surrogate decision
2
The participating mothers were asked several ques-
4
tions related to the issue of surrogate decision. As to the
 U. Gustafsson Stolt et al. / Social Science a Medicine 54 (2002) 1333–1344
Table 4
Factors related to surrogate decision and ethical concerns
Factors related to surrogate decision Number
Category 1Fparticipants in the ABIS project (n ¼ 15)
Awareness of making a surrogate decision (a decision
not only for themselves but also for their child)
Yes 10
No 5
Experience of concern about an ethical issue
Yes 1 about it a lot recently since we now have decided to
No 14
Reasons given for’’ legitimate’’ right to decide
A decision in the child’s best interests 9 wrong! The children do have a right to decide when it
Parental right: ‘‘we decide.’’ 5 concerns them. If they for example can decide if they
(Trying to make) a decision FOR the child – 1 want fishfingers or not, they can decide more
something he/she would have wanted/done
question ‘‘Were you aware of making a decision not
only for yourself but also for your child?’’ 10 answered
that they recall having been. But, as Table 4 shows, 14 of
them did not consider any ethical issues, i.e. did not
claim to have experienced any concern regarding it. The
following quotations illustrate the general opinion:
You decide because you believe it is in the childs best
interests. That is what we are meant to do (Pmo 10,
32:2).
I think it is quite alright to decide about things like
this that will help her in the future. If what we decide
is beneficial to her, why not? (Pmo 11, 36:2).
As an adult you make decisions all the time: I have
decided to have her baptized and that today she will
wear that sweatshirt. As an adult you have to set
rules (Pmo 8, 29:2).
The above quotations also illustrate another point
worth making. There seem to be different attitudes
towards what could be called ‘‘legitimate grounds’’ for
them making decision for their child. The most common
attitude is that the mothers feel they have (and had at
the time of the decision) a right to decide for their child
if it is for the child’s best interests. More than half of the
participating mothers share that opinion. But also
another view was present regarding whether or not to
participate in the project was discussed:
Yes, but I do not feel he has an active part in this
whole,project, really. Where ABIS is concerned I
have total right to decide (Pmo 5, 29:1).
In Table 4, this viewpoint, which 5 of the 15 mothers
share, is listed as ‘‘parental right’’:
It is the right of parents, it is not an intrusion on him
in any way (Pmo 9, 36:1).
1339
The one remaining mother did convey concern about
the issue of making decisions for her child, but this was
not something she was troubled by at that time when she
and her husband made their decision to participate in
the project. It is rather something which has grown and
now become more focused upon since they made the
decision to baptise their son:
I have thought about that, actually I have thought
have him baptised. I am not sure he wants toyit is
just my wish and he has to put up with it, and it is so
important things, do you understand? Then I
thinkFdoes he want this? Every person has a right
to speak their mind and then you have to comply
with it as far as possible, but he can’t do that yet. It is
difficult (Pmo 15, 25:1).
Views concerning delicacy of the information and material
collected
As described above, the participating families
contribute a wide variety of information. There are
blood samples taken at the delivery ward, a first
questionnaire asked to be filled in at the maternity
ward, a diary that ‘‘follows’’ the child during its first
year of life. Additional samples are taken at the yearly
controls in conjunctions with follow-up questionnaires
at certain intervals. The questionnaires consist of
questions related to different aspects of environment,
for example living conditions, occupational factors,
illnesses in the larger family context, psychological
factors and food consumption (see Table 5). Both the
questionnaire and the diary have been designed to cover
the widest range possible of environmental factors.
Thus, the information provided by the families is
substantial. The participating mothers were asked how
they perceived the information and material that
they contributed. It emerged that none (0) of them
thought the information they provided was in any way
sensitive. The initial question for this question area was
‘‘Do you feel you understand what the information will
be used for?’’ and all but one said yes. The common
attitude among them was that this kind of information,
and the amount asked for, was necessary to form the
basis of the further project studies. As one of the
mothers describes it:
The more information about people, how they live,
eat and look upon illnessesFthe more information
to learn, research about, teach and to develop
medicines (Pmo 2, 44:2).
1340 U. Gustafsson Stolt et al. / Social Science a Medicine 54 (2002) 1333–1344
Table 5
Factors related to delicacy of information/samples collected
Questions related to delicacy of Number
information/materials
Category 1Fparticipants in the ABIS project (n ¼ 15)
Feeling that the material they have given
(information through questionnaire(s), diary,
samples collected (blood etc)) is delicate
Yes 0 I trust it is being used in a correct way, I have to do
No 15
Expressed wish for restrictions on the availability of the
information collected
Yes 13
No 2 secrecy. So it should be completely harmless, but then
When asked ‘‘How do you look upon the fact that
information from ABIS will be stored in computers,
analysed in different research projects, etc.?’’, the general
attitude among the participating mothers was positive,
i.e. they felt the information not in any way delicate or a
danger to their integrity:
The material I contribute is not sensitive in any way,
that kind of information is not a cause for concern or
a danger to us. If there had been something of that
kind, I would not have given it (Pmo 9, 36.1).
I do not feel it is dangerous, it is just the kind of
information we give to research. It can not hurt me in
any way (Pmo 14, 41:2).
But at the same time there is an awareness, among
almost half of the mothers, of the amount of informa-
tion they and their family give, and that it thus could be
mishandled or otherwise, have consequences for them
and their family. What the data indicate is that all of
them are positive to the information asked by them of
the ABIS management, to use a phrase from one of the
mothers it is considered ‘‘positive information’’. At the
same time there is an attitude of awareness present in the
discussions with, at least some, mothers. One particular
mother explain her concerns for the possible implica-
tions as to her child and the future:
Yes, I think about this a lot, personal integrity etc,
and I think, like is it really right to provide this kind
of information about him ? There is always a positive
and a negative way of thinking about everythingFthe
ABIS information is positive information. But still I
leave plenty of information about himFhe might not
want that the day he understands what it means. If he
should get ill, then a lot of people will know this,
people who really should not, really. But it does not
matter how many will have access to this, the point is
that it is I who have provided it. But the advantages
outweigh the disadvantages (Pmo 15, 25:1).
When you analyse the statements concerning this
awareness there is a clear relation between the informa-
tion given and how it is and will be used. This seems to
indicate that the information itself is considered positive,
the possible ‘‘danger’’ has nothing to do with the actual
data, but with what can be done with it. When asked the
question how she looked upon the material gathered and
to be analysed, one mother answered:
that, otherwise you could not do the work you
should, not continue with these kinds of projects in
the future, unless you deal with your material under
you never know in this world of computers (Pmo 10,
32:2).
This line of thinking is supported by the fact that 13 of
the 15 participating mothers express a wish for restric-
tions in the ABIS material. The general feeling can be
summarised by the following two quotations:
Yes, I believe there should be restrictions. It is
positive information, but still sensitive. People will
know a lot about him. If he does get ill, a lot of
people are going to know about it, they will know
what will happen to him and what genes he has. But
anyway it is positive (Pmo 15, 25:1).
It must not be abused as far as we are concerned.
You can’t give it out to others, only those that are
authorised to should be able to look at it. Otherwise
there can be danger. But at the same time, when you
do participate in something like this you have to be
open to letting other people have access to the
material, but of course I would then like to be asked.
Those who have access to the material should have a
relation to it (Pmo 6, 37:1).
The core seems to be that there is a wish for the
material to stay ‘‘within the project’’.
Views on issues involving the possible discovery of higher
risk
As also described earlier, one of the future aims is to
find ways to detect children who have or will have a
higher risk of developing diabetes. If such methods and
measures are developed one further aim is to offer those
children prevention of some kind. Issues concerning this
were incorporated into the question themes. It emerged
that at least one of the mothers did not know about this,
she had been given the information that this project
mainly concerned future children and families and that
their participation of her and her child would therefore
not have any direct consequences for her own family (see
Table 6).
 U. Gustafsson Stolt et al. / Social Science a Medicine 54 (2002) 1333–1344
Table 6
Factors related to the possible discovery of their child having a
higher risk of developing Type 1 diabetes and other project
results
Questions related to possible results Number
of the screening
Category 1Fparticipants in the ABIS project (n ¼ 15)
Attitudes about a possible discovery of child
having higher risk ofdeveloping diabetes etc
Positive 14
Negative 1 Anxiety is a notion mentioned in the context of feared
Information desired of possible discovery of higher risk/
project results?
Yes 15
No 0 the participation itself, the worry about material and
I did not know I was going to find out anything
personal, for her, only research for future use. I
haven’t thought about it since I did not know this
could be the case (Pmo 4, 43:2).
When the subject was under discussion, however, all
fifteen (15) mothers displayed a positive attitude towards
any future finding of their child having a higher risk of
developing diabetes:
I would regard it as positive, I would, absolutely. But
if that turns out to be the case we will deal with it
then, it is nothing you can go and be worried about
now. But of course I think about it, but it is safer to
be a part of the project since you have more control
and can find out more easily if they would find him to
have a higher risk (Pmo 15, 25:1).
It seems also be a ground for not experiencing any
concern or feelings of distress/anxiety if told or informed
of a result of this kind:
I think it would really good to know, to find out.
Absolutely, and at that time maybe you have come so
far medically that there is something to give. Then I
would feel much more calmFa relief (Pmo 13, 34:2).
One of the question asked in the context of informa-
tion about a possible discovery of a higher risk factor
was ‘‘Do you feel, as parent, that you have a right to be
informed about possible discoveries of the screening?’’
More than half of the mothers here implicitly mention a
sort of trading thinking, since they contribute with their
materials they feel they have a right to know:
Yes, that is my opinion. If they get all my data and all
my samples and then find something wrong, then I
think I have the right to know (Pmo 11, 36:2).
Yes, I think so, and just because I am a participant. It
is my right if I help, so to speak (Pmo 13, 34:2).
1341
One of the participant mothers also invokes the right
of the child in the same context:
Yes, as the mother of a child who is a part of the
study, of course I want to (be informed). It is his right
(Pmo 14, 41:2).
Discussion
negative consequences of screening procedures
(Hermer!en, 1999; Holtzman, 1988; Shickle a Chad-
wick, 1994). Most likely different problems are implied:
samples collected, stored, the anxiety and possible
stigmatisation in respect of results, anxiety caused by
false positive results (Schulte, 1985; Schulte, a Singal,
1996), distress caused by for example blood sampling
procedures, in the case either of the mothers or of the
child and so forth.
Mentioned in this discussion about screening projects
and their consequences for the individuals partaking in
studies of this kind is that the actual participation can
cause distress and anxiety. Our data from this particular
study shows that the mothers feel neither concern nor
any emotional distress or anxiety in respect of their
participation. On the contrary the mothers have a very
positive opinion both of the study and of their own role.
The majority comment on the importance of the project:
research on children is important, not only to them and
their own child but also to society and the future. All
mothers also feel they themselves are important to the
project, their contributions are important for the
continuation and outcome of the ABIS project. Several
mothers also speak of their interest in research and
health in general, others speak more directly of their
wish to help research finding out the causes of, in this
case, child diabetes. One explanation of this is of course
the augmenting of the ‘‘environmental diseases’’ in
society as a whole. Half of the mothers have a personal
experience of allergy, celiaci, diabetes, rheumatism,
either they have it or a close family member.
The positive attitude in general is most likely an
important factor behind the reasons to participate in the
ABIS study. The most common reason stated (14 of the
15 participating mothers) was a wish to help research. A
little less than half of the 15 mothers also stated as
reasons the personal experience of the diseases involved
and that it could be of general help to their own child
(future medicines, treatments, etc.). Five mentioned the
wish to find out if their own child would be in the risk
zone. The general attitude in this study, is one of
benevolence and fits well with other studies of the
psychology of mothers in clinical research and screening
for Duchenne (Harth, Johnstone, a Thong, 1992;
1342 U. Gustafsson Stolt et al. / Social Science a Medicine 54 (2002) 1333–1344
Bradley et al., 1993, etc.). This is also interesting since it
has been mentioned in the discussion of selective
screening that an individual who makes rational choices
will consent to testing only when they perceive a
favourable risk-benefit ratio (Childress, 1997).
One comment to these results is that during the
retrospective interviews a special focus was laid, by the
participant mothers, upon allergies and diabetes.
At least allergies is commonly (by the public) known
as a ‘‘family disease’’, which could, if it is further
interpreted in a mother–child relationship, also serve as
an important factor behind the prevalent positive
attitude as expressed by the mothers. In a way they, at
the same time as expressing the wish for contributing to
research purposes, also implicitly help their own child or
children. The quantitative study might help to bring
further light into this issuein which we also will further
develop our discussion concerning the complex issue of
information. Diabetes also most likely has a strong
symbolic importance, due to the severity and augment-
ing of the disease.
The most common reason (4 out of 6) stated by the
mothers who chose not to participate in the ABIS study
was the concern for putting the child through frequent
blood testing. Additional two expressed concern that the
project involved some kind of ‘‘experimentation’’ invol-
ving their child. These are concerns that need to be taken
seriouslyandmust be considered in every medical re-
search project. Does, then, the ABIS study involve
frequent blood testing of the child?
The original project guideline involves, apart from the
cordal blood taken immediately after delivery, asking
the parents to consent to three blood samples from the
child, taken when the child is approximately 1, 3 and 5
years of age. What our results indicate is that this issue
needs to be discussed further in the context of
information. As Table 2 indicates, there seems to be
some difficulty as to the actual giving and receiving of
information. Of the participant mothers 9 out of 15 do
not recollect having received any oral information, an
additional six state that they did not remember receiving
the letter of information. Where the nonparticipating
mothers are concerned 5 out of 6 acknowledge retro-
spectively in having received both oral and written
information. A special video was produced and was
intended to be shown to all mothers-to-be, none (0) out
of the 21 interviewed mothers recalls having seen it.
These findings are also interesting in the context of the
issue of informed consent, in several respects. The results
seem to indicate that this is a complicated issue: for
example our findings show clearly that all 21 interviewed
mothers say they recollected receiving sufficient/thor-
ough information that enabled them to make a well-
reasoned decision to participate or not. Further, they all
say they made a full voluntary decision without no
external factor of influence, either by staff or family
members, etc. Only two of the nonparticipating mothers
have mentioned that they were in part influenced by
their husbands’ opinions. Whether this is an indication
of a true informed (refering to Beauchamp a Childress,
1994, Chapter 3) consent or a rationalisation at the time
of the interview is difficult to judge at this point of our
studies. One could argue, however, that if they at that
time experienced the information as sufficient, well
informed, etc., the participants did, as far as they are
concerned, an informed consent.
At the same time we must not overinterpret the
frequent use of recollection: the data indicates that the
information given might have varied in some aspects.
Also the findings of our interview study with the staff
involved in this particular project support this to some
extent (Gustafsson-Stolt et al., 2001).
As described above, the possible distress and anxiety
of participants in screening projects can be caused by for
example the (sometimes very personal) information they
give about themselves and by possible results of the
project. That this is a very important issue can be
exemplified by the fact that the ALSPAC study
(mentioned in the introduction) and the ABIS study
have chosen two different standpoints. The principle of
respect for persons, including respecting every indivi-
dual’s integrity, autonomy, right to know and decide
and so forth, provides the ethical foundation of the
decisions made by project management and ethical
committees involved in projects of this magnitude. The
ALSPAC choose anonymity as the way to protect these
rights and the ABIS project has chosen, as an aim, an
attempt to identify individuals with a higher risk of
developing child diabetes and, if possible, to offer
preventive measures to these children. Through their
experiences over the years the ethical committe of the
ALSPAC has seen it necessary to bring up these issues
and certain revisions have been made (Mumford,
1999a, b).
Our findings indicate that the feared negative con-
sequences regarding these issues might be unwarranted
in some cases. The general attitude amongst the mothers
in this study is also positive on this point. When asked
questions about how they perceive, in general, the
material they contribute the ABIS project (information
through questionnaires diary, different samples taken)
none of them say they experience it as sensitive or
delicate information. On the contrary, the importance
and necessity of the material for the project’s outcome
and results is stressed by the majority of the mothers.
They also did not feel that the material in question
constituted the grounds for concern or a potential cause
of harm, either to them or their child. One could argue,
however, that the statements made by the respondents
concerning the wish for restrictions of the blood samples
taken is an indication of another opinion, i.e. an
experience of the delicacy of the information, regarding
 U. Gustafsson Stolt et al. / Social Science a Medicine 54 (2002) 1333–1344
at least these samples. The written information was not
in the same way conceived as delicate, supported by the
fact that it was not mentioned in the discussion about
restrictions.
When asked how they feel about any positive results,
i.e. if their child should be considered to have a higher
risk of developing child diabetes, they all show a
favourable attitude. Instead of being a cause for concern
or anxiety they feel it would be a positive thing to find
out. Since they are part of the project the child can get
help, preventive measures, etc., ‘‘its better to find out
early’’. And, as Table 5 and subsequent quotations
clearly show, they all feel its their right to be informed of
any positive results, both as participants and as parents.
There was also a stress on the duty of the project
management to inform them when asked how they
perceived the projects management duties to the
participant families, not only of any kind of future
results but also of the progress of the project.
Even though the interviewed participant mothers did
not experience any concern regarding the material
gathered about them and their family, they all expressed
a clear wish for restrictions of the material: how it is
used, by whom, etc. Several used the phrase ‘‘positive
information’’ about the ABIS information but the
statements at the same time indicate an awareness of
the potential danger of misuse. A common wish
expressed with regard to this particular question area
was for the material to be ‘‘kept in the project’’ and
‘‘used for ABIS projects’’. We are aware that this
question is not unrelated to the issue of information, but
the statements indicate at least a certain level of
knowledge/understanding of the ABIS projects and
aims.
One last issue incorporated into the interviews was
surrogate decision. The results of this study show that
even though a clear majority of the respondents said
they were, both at that time of the decision and during
the interview, aware of making a surrogate decision for
their child, they did not experience the situation as
ethically problematic. When asked how they thought
about a question like this, the overall attitude was one of
stressing the right and necessity of parents to decide for
their child/children.
Conclusion
Large-scale screening projects may cause concern or
anxiety and distress and thereby a negative attitude on
the part of the participants. Our empirical study
suggests, however, that mothers with very different
background (age, education, parity) display a positive
attitude to this particular neonatal research screening, in
this case represented by the ABIS project (All Babies in
Southeast Sweden). The majority of the respondents do
1343
not report any concern regarding issues of for example
their participation, most of the material collected for
project purposes, surrogate decision the question of
project potential results. The ethical problems put
forward in more theoretical contexts is not, at large,
supported by this explorative interview study.
One conclusion is therefore that the feared grounds
for concern might be unwarranted in those cases where
there seem to be a firm public support and interest for
the project purpose. Maybe one can argue for a more
differentiated discussion regarding different kinds of
screening.
The basis for the positive attitude could be the
commonly expressed wish of helping research and the
possibility of the particular research to be of help to
their own child and others. The importance of research
about allergies, diabetes, celiaci is also stressed by the
mothers. On the other hand, the participants expressed
concern about the storage and use of material (and
possible infringement of) and also the right to be told of
any results of the screening. The study also displays the
importance of finding adequate ways of conveying
information to all categories involved. This study clearly
conveys the complexity of issues in any discussion of
screening and its ethical implications, both as partici-
pants, research management and ethical decision ma-
king.The study also serves as an empirically grounded
example of issues that needs to be investigated into
further, mainly in the context of information.
Acknowledgements
The authors would first of all like to thank all the
mothers and staff who have participated in this study.
We are also grateful for the help received from the staff
of the Maternity Units, the ABIS nurses Caroline B,
Christina L and Iris F and to the administrative staff
Ann-Christine Gilmore-Ellis and Annelie Wass. This
research has been financially supported by the Juvenile
Diabetes Foundation (JDF) - Wallenberg no K98-99JD-
12813-01A, the Swedish Child Diabetes Foundation
(Barndiabetesfonden) and Torsten and Ragnar
So. derbergs Foundation.
References
Beauchamp, T. L. (1997). Informed consent. In R. M. Veatch
(Ed.), Medical ethics (2nd ed.). Sudbury: Jones and Bartlett
Publishers.
Beauchamp, T. L., a Childress, J. F. (1994). Principles of
biomedical ethics (4th ed.). New York: Oxford University
Press.
Bradley, D. M., Parsons, E. P., a Clarke, A. J. (1993).
Experience with screening newborns for Duchenne muscular
1344 U. Gustafsson Stolt et al. / Social Science a Medicine 54 (2002) 1333–1344
dystrophy in Wales. British Medical Journal, 306(6874),
357–360.
Brody, H. (1997). The physician-patient relationship. In R. M.
Veatch (Ed.), Medical ethics (2nd ed.). Sudbury: Jones and
Bartlett Publishers.
Childress, J. F. (1997). Practical reasoning in bioethics.
Bloomington and Indianapolis: Indiana University Press.
Faden, R. R., Holtzman, N. A., a Chwalow, A. J. (1982).
Parental rights, child welfare, and a public health: The case
of PKU screening. American Journal of Public Health,
72(12), 1396–1400.
Faden, R. R., Chwalow, A. J., Orel-crosby, E., Holtzman,
N. A., Chase, G. A., a Leonard, C. O. (1985). What
participants understand about a maternal serum alpha-
fetoprotein screening program. American Journal of Public
Health, 75(12), 1381–1384.
Fenton-May, J., Bradley, D. M., Sibert, J. R., Smith, R.,
Parsons, E. P., Harpes, P. S., a Clarke, A. (1994).
Screening for Duchenne muscular dystrophy. Archives of
Diseases in Childhood, 70(6), 551–552.
Fost, N. (1999). Ethical implications of screening asymptomatic
individuals. In Beauchamp, D. E., a Steinbock, B. (Eds.),
New ethics for the Public’s Health. New York: Oxford
University Press.
Golding, J. (1989). European longitudinal study of pregnancy
and childhood (ELSPAC). Paediatric and perinatal epidemiol-
ogy, 3, 460–469 (In Mumford, S.E. stated as page 279–293).
Golding, J., a The ALSPAC Study Team. (1996). Children of
the nineties: A resource for assessing the magnitude of long-
term effects of prenatal and perinatal events. Contemporary
review in obstetrics and gynaecology, 8, 89–92.
Gustafsson Stolt, U., Liss, P.-E., a Ludvigsson, J. (2001).
Perspectives of bioethics: An interview study of health care
professionals involved in a research screening project of
children, in press.
Harth, S. C., Johnstone, R. R., a Thong, Y. H. (1992). The
psychological profile of parents who volunteer their children
for clinical research: A controlled study. Journal of Medical
Ethics, 18, 86–93.
Hermer!en, G. (1999). Neonatal screening: Ethical aspects. Acta
Paediatrica Supplement, 432, 99–103.
Holm, S. (1996). Ethical problems in clinical practice–a study of
the ethical reasoning of health care professionals. Disserta-
tion, University of Copenhagen.
Holtzman, N. A. (1988). Recombinant DNA technology.
Genetic tests and public policy. American Journal of Human
Genetics, 42, 642.
Holtzman, N. A., Faden, R., Chwalow, A. J., a Horn, S. D.
(1983). Effect of informed parental consent on mothers’
knowledge of newborn screening. Paediatrics, 72(6),
807–812.
Kvale, S. (1997). Den kvalitativa forskningsintervjun. Lund:
Studentlitteratur.
Mumford, S. E. (1999a). Children of the 90s: Ethical guidance
for a longitudinal study. Archives of Diseases in Childhood
Neonatal Education, 81, 146–151.
Mumford, S. E. (1999b). Children of the 90s II: Challenges for
the ethics and law committee. Archives of Diseases in
Childhood Neonatal Education, 81(3), 228–231.
Parsons, E., Bradley, D., a Clarke, A. (1996). Disclosure of
Duchenne muscular dystrophy after newborn screening.
Archives of Diseases in Childhood, 74, 550–553.
Patton, M. Q. (1980). Qualitative Evaluation Methods (6th ed.).
Thousand Oaks: Sage Publications.
Riis, P. (1999). Ethical, legal and health economic aspects of
neonatal screening. Acta Paediatrica Supplement, 432,
96–98.
Schulte, P. A. (1985). The epidemiologic basis for the
notification of subjects to cohort studies. American Journal
of Epidemiology, 121, 351–361.
Schulte, P. A., a Singal, M. (1996). Ethical issues in the
interaction with subjects and disclosure of results. In S.
Coughlin, a T. Beauchamp (Eds.), Ethics and epidemiology
(pp. 178–196). Oxford: Oxford University Press.
Shickle, D., a Chadwick, R. (1994). The ethics of screening: Is
screeningitis an incurable disease? Journal of Medical Ethics,
20, 12–18.
Smith, R. A., Williams, D. K., Sibert, J. R., a Harper, P. S.
(1990). Attitudes of mothers to neonatal screening for
Duchenne muscular dystrophy. British Medical Journal,
300(6732), 1112.
Trost, J. (1997). Kvalitativa intervjuer: Andra upplagan. Lund:
Studentlitteratur.
Veatch, R. M. (1989). Medical ethics ((2nd ed.)). Sudbury:
Jones and Bartlett Publishers.
Walters, L. (1997). Reproductive technologies and genetics. In
R. M. Veatch (Ed.), Medical Ethics (2nd ed.). Sudbury:
Jones and Bartlett Publishers.