Obstacles to conducting epidemiological research in the UK general population
http://www.100md.com
《英国医生杂志》
1 National Creutzfeldt-Jakob Disease Surveillance Unit, Edinburgh EH4 2XU, 2 London School of Hygiene and Tropical Medicine, London WC1E 7HT
Correspondence to: H J T Ward h.ward@ed.ac.uk
Experiences from a national case-control study of Creutzfeldt-Jakob disease show the tensions between protecting individual patients' confidentiality and the access required for the benefit of public health
Introduction
Advances in computer technology have given rise to fears about access to patients' records. The UK response was to supplement doctors' common law duty of confidentiality to their patients with the Data Protection Act 1998. Much debate has ensued about the extent to which medical research could be impeded, including issues surrounding informed consent, patient confidentiality, anonymisation, and access to data.1 2 The subsequent updating of guidance on confidentiality by various professional organisations has produced inconsistencies, adding to the confusion about what constitutes ethically acceptable research.3-5 Fears have been expressed that epidemiological research will be severely hampered, and disease surveillance was severely threatened in England and Wales before the introduction of section 60 of the Health and Social Care Act 2001.6
UK case-control study of Creutzfeldt-Jakob disease
With the Data Protection Act 1998 in its infancy and uncertainty surrounding its interpretation, there was concern about how the study could be carried out without breaching patient confidentiality. We submitted an application to a multicentre research ethics committee in July 1998 and obtained approval, after some amendments, in October 1998. Under the revised protocol, we would write to the general practitioners of patients with Creutzfeldt-Jakob disease and ask them to take part in the study. If they agreed, the general practitioners would be asked to write (using pre-prepared letters) to 20 people of similar age and sex to the case, randomly selected from their patient database. These individuals were asked to consent to be contacted by the National Creutzfeldt-Jakob Disease Surveillance Unit. We then wrote to those who consented, inviting them to participate in the study and to nominate a relative to be interviewed. (Because cases are typically too unwell to participate, a relative is interviewed, and so we had to do the same for controls.) The relatives of the controls who agreed were approached, and if they also consented, a nurse practitioner visited and interviewed them at a convenient location.
As a condition of our ethical approval, we also had to approach 213 local research ethics committees. This was because at that time no provision was made for studies that did not have local researchers. The task was cumbersome, partly because there was no uniformity in what was required by each local ethics committee or in how they discharged their responsibility.2 8-10 Since 2000, studies with no local researcher and that meet certain conditions are not required to seek approval from local research ethics committees, although the above concerns still apply to studies with local investigators.
Recruiting controls
It is now difficult to recruit community based controls from general practices without introducing scope for a substantial degree of selection bias, partly because of the status of patient confidentiality in Europe and the United Kingdom. In our study, an additional barrier was created because we could not approach people unless they had sent written consent to their general practitioner. We believe this was partly responsible for the poor initial response rate of 37%.
Another problem was that the ethical approval required general practitioners to send detailed written information about the study. An important part of the process of recruiting people into studies is explaining to them why the study is important and what is being asked of them. This is particularly true when the study is as complicated as ours. We believe that relying on letters, with no early opportunity for the researchers to interact directly with potential participants, contributed to the low response rate.
Our study has an excess of healthcare workers in the control group, which may bias our results. Because of the potential for selection bias resulting from the low response rate, and the complicated logistics involved, we have had to explore other approaches to recruiting controls.
Balancing costs to the individual with benefits to society
Alternative approaches to recruiting community controls include using publicly available databases, picking random houses in a street, or asking cases (or their relatives) to nominate a relative or friend. Each of these has advantages and disadvantages. The use of publicly available databases—for example, telephone directory listings or the postcode address file, raises an interesting issue. People on these databases can be (and are) approached by researchers or salespeople without any ethical approval, although research funded within the health service will usually require that ethical approval is obtained. However, health researchers are not permitted to contact directly individuals who have not replied to a letter from their general practitioner because their name has been obtained from a general practice list.
Summary points
Issues of patient confidentiality are hampering epidemiological research
Wider debate is required about the use of medical records to identify eligible individuals
Ethics committees should weigh the benefits to society against costs to the individual when considering studies
Use of public funds should be considered as part of the cost to society if a study cannot recruit participants by the most effective method
If we had contacted individuals who had not responded to a letter from their general practitioner (to which they could have responded indicating that they did not wish to be contacted) would this have constituted an unacceptable breach of patient confidentiality or an unacceptable infringement of their privacy? Further debate is needed on how to strike an appropriate balance between an individual's rights and freedoms and the right of the community or society to answer important questions. Perhaps patients of general practitioners should have the opportunity to opt out of being contacted for health research purposes in the same way as some private companies offer customers the opportunity to indicate that they do not want their names passed on to other interested parties.
Editorial by Warlow
This study would not have been possible without the cooperation of the relatives of patients with Creutzfeldt-Jakob disease and controls and their relatives. We thank research staff at the unit for collecting data (M Zeidler, G Stewart, M A Macleod, C Henry, A Lowman, S Cooper, C Heath, K Murray) and N Attwood for database management.
Contributors and sources: HJTW is an epidemiologist and public health specialist. BSB and ML are research nurses who visit and interview the relatives of controls in the study. SNC and PGS have been involved in epidemiological research into Creutzfeldt-Jakob disease for many years. DE is a medical statistician. RGW set up the surveillance unit in 1990 and was its director for many years. The article arose from a number of discussions around the problem of the low response rate. HJTW analysed the data and prepared the first draft of the manuscript. DE, SNC, PGS, RGW reviewed the data. All authors contributed to writing and redrafting the article. HJTW is the guarantor.
Funding: The National CJD Surveillance Unit is funded by the Department of Health and the Scottish Executive Health Department. The case-control study investigating risk factors for sporadic CJD is funded by the Department of Health (DH 121/6407 and 121/7400). The views expressed are those of the authors.
Competing interests: None declared.
References
Strobl J, Cave E, Walley T. Data protection legislation: interpretation and barriers to research. BMJ 2000;321: 890-2.
Al-Shahi R, Warlow C. Using patient-identifiable data for observational research and audit. BMJ 2000;321: 1031-2.
General Medical Council. Confidentiality. London: GMC, 2000.
British Medical Association. Confidentiality and disclosure of health information. London: BMA, 1999.
Medical Research Council. Professional information in medical research. London: MRC, 2000.
Health and Social Care Act 2001. London: Stationery Office, 2001.
Will RG. Surveillance of prion diseases in humans. In: Baker HF, Ridley RM, eds. Methods in molecular medicine: prion diseases. Totowa, NJ: Humana Press, 1996: 119-37.
Alberti KGMM. Multicentre research ethics committees: has the cure been worse than the disease. BMJ 2000;320: 1157-8.
Tully J, Ninis N, Booy R, Viner R. The new system of review by multicentre research ethics committees: prospective study. BMJ 2000;320: 1179-82.
Lux AL, Edwards SW, Osborne JP. Responses of local research ethics committees to a study with multicentre research ethics committee approval. BMJ 2000;320: 1182-3.(Hester J T Ward, consulta)
Correspondence to: H J T Ward h.ward@ed.ac.uk
Experiences from a national case-control study of Creutzfeldt-Jakob disease show the tensions between protecting individual patients' confidentiality and the access required for the benefit of public health
Introduction
Advances in computer technology have given rise to fears about access to patients' records. The UK response was to supplement doctors' common law duty of confidentiality to their patients with the Data Protection Act 1998. Much debate has ensued about the extent to which medical research could be impeded, including issues surrounding informed consent, patient confidentiality, anonymisation, and access to data.1 2 The subsequent updating of guidance on confidentiality by various professional organisations has produced inconsistencies, adding to the confusion about what constitutes ethically acceptable research.3-5 Fears have been expressed that epidemiological research will be severely hampered, and disease surveillance was severely threatened in England and Wales before the introduction of section 60 of the Health and Social Care Act 2001.6
UK case-control study of Creutzfeldt-Jakob disease
With the Data Protection Act 1998 in its infancy and uncertainty surrounding its interpretation, there was concern about how the study could be carried out without breaching patient confidentiality. We submitted an application to a multicentre research ethics committee in July 1998 and obtained approval, after some amendments, in October 1998. Under the revised protocol, we would write to the general practitioners of patients with Creutzfeldt-Jakob disease and ask them to take part in the study. If they agreed, the general practitioners would be asked to write (using pre-prepared letters) to 20 people of similar age and sex to the case, randomly selected from their patient database. These individuals were asked to consent to be contacted by the National Creutzfeldt-Jakob Disease Surveillance Unit. We then wrote to those who consented, inviting them to participate in the study and to nominate a relative to be interviewed. (Because cases are typically too unwell to participate, a relative is interviewed, and so we had to do the same for controls.) The relatives of the controls who agreed were approached, and if they also consented, a nurse practitioner visited and interviewed them at a convenient location.
As a condition of our ethical approval, we also had to approach 213 local research ethics committees. This was because at that time no provision was made for studies that did not have local researchers. The task was cumbersome, partly because there was no uniformity in what was required by each local ethics committee or in how they discharged their responsibility.2 8-10 Since 2000, studies with no local researcher and that meet certain conditions are not required to seek approval from local research ethics committees, although the above concerns still apply to studies with local investigators.
Recruiting controls
It is now difficult to recruit community based controls from general practices without introducing scope for a substantial degree of selection bias, partly because of the status of patient confidentiality in Europe and the United Kingdom. In our study, an additional barrier was created because we could not approach people unless they had sent written consent to their general practitioner. We believe this was partly responsible for the poor initial response rate of 37%.
Another problem was that the ethical approval required general practitioners to send detailed written information about the study. An important part of the process of recruiting people into studies is explaining to them why the study is important and what is being asked of them. This is particularly true when the study is as complicated as ours. We believe that relying on letters, with no early opportunity for the researchers to interact directly with potential participants, contributed to the low response rate.
Our study has an excess of healthcare workers in the control group, which may bias our results. Because of the potential for selection bias resulting from the low response rate, and the complicated logistics involved, we have had to explore other approaches to recruiting controls.
Balancing costs to the individual with benefits to society
Alternative approaches to recruiting community controls include using publicly available databases, picking random houses in a street, or asking cases (or their relatives) to nominate a relative or friend. Each of these has advantages and disadvantages. The use of publicly available databases—for example, telephone directory listings or the postcode address file, raises an interesting issue. People on these databases can be (and are) approached by researchers or salespeople without any ethical approval, although research funded within the health service will usually require that ethical approval is obtained. However, health researchers are not permitted to contact directly individuals who have not replied to a letter from their general practitioner because their name has been obtained from a general practice list.
Summary points
Issues of patient confidentiality are hampering epidemiological research
Wider debate is required about the use of medical records to identify eligible individuals
Ethics committees should weigh the benefits to society against costs to the individual when considering studies
Use of public funds should be considered as part of the cost to society if a study cannot recruit participants by the most effective method
If we had contacted individuals who had not responded to a letter from their general practitioner (to which they could have responded indicating that they did not wish to be contacted) would this have constituted an unacceptable breach of patient confidentiality or an unacceptable infringement of their privacy? Further debate is needed on how to strike an appropriate balance between an individual's rights and freedoms and the right of the community or society to answer important questions. Perhaps patients of general practitioners should have the opportunity to opt out of being contacted for health research purposes in the same way as some private companies offer customers the opportunity to indicate that they do not want their names passed on to other interested parties.
Editorial by Warlow
This study would not have been possible without the cooperation of the relatives of patients with Creutzfeldt-Jakob disease and controls and their relatives. We thank research staff at the unit for collecting data (M Zeidler, G Stewart, M A Macleod, C Henry, A Lowman, S Cooper, C Heath, K Murray) and N Attwood for database management.
Contributors and sources: HJTW is an epidemiologist and public health specialist. BSB and ML are research nurses who visit and interview the relatives of controls in the study. SNC and PGS have been involved in epidemiological research into Creutzfeldt-Jakob disease for many years. DE is a medical statistician. RGW set up the surveillance unit in 1990 and was its director for many years. The article arose from a number of discussions around the problem of the low response rate. HJTW analysed the data and prepared the first draft of the manuscript. DE, SNC, PGS, RGW reviewed the data. All authors contributed to writing and redrafting the article. HJTW is the guarantor.
Funding: The National CJD Surveillance Unit is funded by the Department of Health and the Scottish Executive Health Department. The case-control study investigating risk factors for sporadic CJD is funded by the Department of Health (DH 121/6407 and 121/7400). The views expressed are those of the authors.
Competing interests: None declared.
References
Strobl J, Cave E, Walley T. Data protection legislation: interpretation and barriers to research. BMJ 2000;321: 890-2.
Al-Shahi R, Warlow C. Using patient-identifiable data for observational research and audit. BMJ 2000;321: 1031-2.
General Medical Council. Confidentiality. London: GMC, 2000.
British Medical Association. Confidentiality and disclosure of health information. London: BMA, 1999.
Medical Research Council. Professional information in medical research. London: MRC, 2000.
Health and Social Care Act 2001. London: Stationery Office, 2001.
Will RG. Surveillance of prion diseases in humans. In: Baker HF, Ridley RM, eds. Methods in molecular medicine: prion diseases. Totowa, NJ: Humana Press, 1996: 119-37.
Alberti KGMM. Multicentre research ethics committees: has the cure been worse than the disease. BMJ 2000;320: 1157-8.
Tully J, Ninis N, Booy R, Viner R. The new system of review by multicentre research ethics committees: prospective study. BMJ 2000;320: 1179-82.
Lux AL, Edwards SW, Osborne JP. Responses of local research ethics committees to a study with multicentre research ethics committee approval. BMJ 2000;320: 1182-3.(Hester J T Ward, consulta)