Attitudes to bioethical issues: a case study of a screening project

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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 benevolence—they 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.

Introduction

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, & Chwalow 1982; Holtzman, Faden, Chwalow, & Horn, 1983; Faden et al., 1985; Bradley, Parsons, & Clarke, 1993; Parsons, Bradley, & 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 Longitudinal Study of Pregnancy and Childhood) and ABIS (All Babies in Southeast Sweden). Even though they differ on some points, for example in questions of 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 & 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 & Singal, 1996; Holtzman, 1988; Hermerén, 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 (Hermerén, 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, & 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 important issue of informed consent, there is limited data concerning actual consent practices, with the result that guidelines, codes, etc. depend heavily on theory (Beauchamp, 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).

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 autoimmune 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, vaccinations, 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.

The objective of this paper is to explore and describe ethically relevant opinions and attitudes among a sample of participants and nonparticipants in respect of the ABIS project.

Section snippets

Participants

The sample for our qualitative study consisted of 21 women who had just become mothers, 15 of whom consented to participate in the project, which reflects the proportion of mothers who accepted or declined to participate in the main study. We did not choose a representative material, but made a strategic selection concerning both categories of respondents. The variables age, education, marital status and parity were utilised to reflect a broad range of different types of mothers with different

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 successful—then it is important and good. It is also very good for my own sake, for example it will help me to see the connection

Discussion

Anxiety is a notion mentioned in the context of feared negative consequences of screening procedures (Hermerén, 1999; Holtzman, 1988; Shickle & Chadwick, 1994). Most likely different problems are implied: the participation itself, the worry about material and samples collected, stored, the anxiety and possible stigmatisation in respect of results, anxiety caused by false positive results (Schulte, 1985; Schulte, & Singal, 1996), distress caused by for example blood sampling procedures, in the

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 not report any concern regarding issues of for example their

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

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