Disease Registers in England - CiteSeerX

Disease Registers in England A report commissioned by the Department of Health Policy Research Programme in support of the White Paper entitled Saving Lives: Our Healthier Nation

John Newton and Sarah Garner

Published by the Institute of Health Sciences University of Oxford Old Road, Oxford OX3 7LF ISBN 1 8407 50286 February 2002

Picture: Wellcome Library, London

Aunt Sophy. “Now suppose, George, as a single woman I should have my name put on the Register, what should I get by it?” Pet Nephew. “Oh, a good deal. You’d be allowed to serve on Coroner Juries, Common Juries, Annoyance Juries, pay Powder Tax and Armorial Bearings, act as Parish Beadle and Night Constable of the casual Ward, and Inspector of Nuisances, report on Fever Districts, and all jolly things of that sort.”

“In considering the management of patients with chronic diseases, an accurate well-maintained register is a prerequisite to providing comprehensive and co-ordinated care.” Cheales & Howitt, 19961

“Without more widespread use of disease registers it is difficult to see how health authorities can meet their responsibility to assess their populations’ needs.” Donaldson, 19922

“Where is the wisdom we have lost in knowledge? Where is the knowledge we have lost in information?” T.S.Eliot

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Acknowledgements We are very grateful to all those who have provided information and ideas for this report, in particular those responsible for running disease registers, the respondents to our survey, the Public Health Observatories and our contacts in the Department of Health and at the MRC. The views expressed in this report are the authors’ own and do not necessarily reflect the policy of the Department of Health.

Contact us at: Institute of Health Sciences, Old Road, Oxford, OX3 7LF [email protected]

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CONTENTS SUMMARY . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . 1 RECOMMENDATIONS. . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . 5 BACKGROUND Context . . . . Remit . . . . . . Approach . . .

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SOME GENERIC ISSUES IN RELATION TO DISEASE REGISTERS . . . . . . . . . . . . . . . . . . 8 Definitions and Terminology . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . 8 Historical Development of Registers . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . 13 Current Uses of Registers . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . 14 The Characteristics of Successful Registers . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . 20 How to Set Up a Disease Register. . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . 22 Evaluating the Quality and Value of Registers . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . 27 CONFIDENTIALITY AND DATA PROTECTION . . . . . . . . . . . . . . . . . . . . Medical Research Council Guidance . . . . . . . . . . . . . . . . . . . . . . . . . . . . The Data Protection Act 1998 . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . Human Rights Act 1998 . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . Common Law Considerations . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . General Medical Council Guidance. . . . . . . . . . . . . . . . . . . . . . . . . . . . . NHS Policy on Confidentiality . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . Health and Social Care Act 2001 . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . Why Do Registers Need Identifiable Data? . . . . . . . . . . . . . . . . . . . . . . . Why Can’t Registers Always Obtain Consent Before Using Personal Data?

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SOME KEY QUESTIONS IN RELATION TO DISEASE REGISTERS . . . . . . Can the Same Registers be Used to Support Patient Care and Research? Are Registers an Alternative to RCTs? . . . . . . . . . . . . . . . . . . . . . . . . . . How Much do Registers Cost? . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . How Should Registers be Funded? . . . . . . . . . . . . . . . . . . . . . . . . . . . . What is the National Role in Respect of Registers?. . . . . . . . . . . . . . . . .

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RESULTS OF THE NATIONAL SURVEY OF DIRECTORS OF PUBLIC HEALTH . . . . . . . 53 Views of Directors of Public Health . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . 53 Views of Regional Directors of R&D . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . 58 REFERENCES . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . 60 APPENDIX APPENDIX APPENDIX APPENDIX APPENDIX

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STRUCTURE OF OUR DATABASE OF REGISTERS BUDGET FOR A TYPICAL REGIONAL REGISTER DIABETES REGISTERS DEVICE REGISTERS SUMMARY OF REGISTERS IDENTIFIED IN THE SURVEY

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Summary

Disease Registers in England

SUMMARY 1.

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Registers have a significant record of contributing to medical knowledge and health care, particularly in the UK. However, they can be expensive and require ethical justification because of concern for civil liberties. It is important to reflect carefully and objectively on their role in a modern health service. Current government policy firmly supports the use of disease registers to improve care and prevent ill health. In the White Paper Saving Lives: Our Healthier Nation there is a commitment to “… strengthen the information base on chronic diseases in the population by establishing a series of disease registers in different parts of the country” (paragraph 11.33). The NHS Plan states that “… primary care trusts will identify and maintain registers of those at greatest risk from serious illness—concentrating particularly on areas where ill health is most prevalent—so that people can be offered preventive treatment” (paragraph 1.6). In the National Service Framework (NSF) for coronary heart disease (CHD) primary care teams are expected to establish and use registers of patients at risk of CHD. The Department of Health commissioned this report in March 2000. We were asked to: (a) outline the potential roles of disease- or condition-based registers for clinical, public health and research purposes; (b) identify what is already being done, which registers have been used; what makes a “useful” register; and how much they cost; (c) indicate how a system of registers might be co-ordinated regionally and nationally. We reviewed the literature, wrote to Regional Directors of Research and Development, Regional Directors of Public Health and District Directors of Public Health. We contacted 117 specific informants in relation to individual registers. In this report we present the general findings. An account of lessons from individual registers will be produced separately.

Main findings 6. There is a substantial and useful literature relevant to disease registers but which is relatively inaccessible. There are helpful discrete literatures on CHD registers, diabetes registers and cancer registers. Inconsistent terminology is often a barrier in searching the literature. 7. The number of disease registers already in existence in England is large, possibly larger than is generally appreciated. Our review was not exhaustive: even so we identified about 250 registers. We would not be surprised if there were more than 400 specific registers in existence in England (excluding CHD and diabetes registers in general practice). 8. There is heterogeneity in every aspect of these registers (size, quality, purpose, topic, cost, funding source, etc.). This heterogeneity reflects the fragmentation of policy and lack of strategy in this area. Cancer registries are the exception to this rule because there is now a developing national framework and regional structures to support it.

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Summary

We reserve the terms “disease register” and “case register” for those databases that attempt to identify all cases of a disease or condition in an identified denominator population. Such registers can be used for epidemiological research and needs assessment, as well as to improve clinical care, and service quality and to perform technology assessment. Clinical databases do not (necessarily) relate to a defined denominator population and usually draw cases from those treated in a particular institution or group of institutions. The main objectives in the use of clinical databases are quality improvement and health technology assessment. For these purposes, a defined denominator is not necessarily required. The main uses of registers are: (a) patient care (regular review and recall, structured care programmes, monitoring high risk groups, managing demand and regulating access, communication, risk stratification); (b) public health (surveillance, planning the provision of health care, monitoring the burden of ill health in the population, monitoring the impact of preventive measures); (c) technology assessment; (d) research (descriptive studies, improving the performance of other research designs, studies of process, hypothesis testing). Requirements for a successful register include an appropriate multidisciplinary team, stable funding, focused aims, data collection systems and design that relate well to function and relevant leadership. Before setting up a register an Expert Group and a Steering Group should be convened. The Information Commissioner should be notified and Research Ethics Committee approval obtained if required. Arrangements for access to the data, data security, accountability, reporting and publicity should then be established. A robust and appropriate case definition is crucial. Methods of ascertaining cases in an unbiased fashion need to be carefully considered. Procedures for identifying duplicates and for follow up may also be important. Evaluation of a register should include: an assessment of the public health importance of the topic, a description of the systems used and their objectives, a description of the uses and other outputs of the register, an evaluation of the system against a set of technical attributes, a description of the resources used to operate the system and a summary of whether the system is meeting its objectives and any modifications required. Disease registers must comply with current legislation on data protection and confidentiality. In particular, if identifiable data are used the patients’ consent to inclusion on the register should be obtained unless there is a good reason not to do so. Consent or refusal should be noted in the medical record and should be respected unless overridden by the law. Consent is already obtained for many registers although much more work is required in this area. Nevertheless, there are cases where it is impractical or impossible to obtain consent from all subjects. There is also empirical evidence that the obligation to obtain express consent results in levels of bias that could invalidate epidemiological research.

Summary

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Registers can rarely be operated using fully anonymised data. There is usually a need to “identify” the individuals concerned, although not necessarily by name, for the purpose of record-linkage. This is for four main reasons: to avoid doublecounting; to construct longitudinal records for the determination of cause and effect; to validate the register against external datasets; to make linkages with other unrelated datasets to test research hypotheses. The NHS number carries the prospect of an important benefit for registers allowing them to operate to a much greater extent using linked anonymised data. The NHS number can be encrypted for further security. We welcome efforts by the Department of Health to encourage use of the NHS number more widely. However, the NHS number is not completely secure, is not generally available on historical records, and is not on all current records of interest. A register using the NHS number or some other form of linked anonymised data (where the register also holds the key) would still be deemed in law to be using identifiable data. A register using such data without obtaining explicit consent can comply with the Data Protection Act 1998 provided: (a) patients in the denominator population are aware in general terms that health data held about them may be collated by central registers for patient care, healthcare management and research without their explicit consent; (b) cogent reasons exist for not obtaining explicit consent (either practical or methodological) and for requiring identifiable data; (c) the work of the register can be shown to be necessary for a legitimate medical purpose that could not be accomplished by some other means; (d) the data controller, and anybody else to whom the data are disclosed, is a medical practitioner or other professional who owes an equivalent duty of confidentiality to the patient. Research registers are exempt from some further provisions of the Act if, among other conditions, the data are used only for research, the patients’ care is not influenced in any way by the processing of those data, and the patient is not contacted by anyone other than their normal professional carer. Some uses of data that are legal under the Data Protection Act 1998 remain a breach of confidentiality under common law. Important new legislation has been incorporated into the Health and Social Care Act 2001 allowing the Secretary of State to make regulations covering the processing of patient information for medical purposes, where this might otherwise be subject to legal challenge under common law. These regulations are very important for disease registers. In future there will be only two classes of disease register using identifiable data: (a) those where patients may need to be contacted directly for clinical purposes – these will almost always require patient consent; (b) those where it is impracticable to obtain consent – these will only be legal if covered by regulations drafted under Section 60 of the Health and Social Care Act. A good quality register generally requires a dedicated administrator and therefore has a minimum cost of around £30,000 per annum. Most modest Regional or District registers cost between £50k and £80k to perform the central functions or the register. Size, in terms of numbers of cases registered (or screened), partly determines cost, as does quality. An economy of scale would seem to

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Summary

apply to registers in that regional registers often do not cost much more than district ones. Some costs will be incurred in the sites reporting data but these are not normally included in the budget for the register. Those responsible for registers are concerned about the future. Lack of a rational mechanism for the funding of disease registers was a common complaint in our survey. Many registries were surviving on small grants from a variety of sources. Much time and energy were diverted into efforts to raise money rather than run the registers and report the results. It is also difficult to establish which agency is responsible for the support or quality assurance of registers. The changing data protection environment and uncertainty over the legality of current practices were also perceived to be threats to the future of many registers. A devolved approach to disease registers can be helpful when responding to local interests and needs. However, current arrangements suffer from a lack of central co-ordination and a lack of consistency in policy. If the Government’s stated aims are to be achieved, there is a need to develop and apply a strategy for registers and to set and monitor standards nationally.

Recommendations


RECOMMENDATIONS I.

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A national strategy for disease registers should be formulated in which the four different purposes of registers (patient care, public health, technology assessment, and research) are recognised. The strategy should include a system for appraising individual registers against agreed standards and for making rational resource allocation decisions. Public health observatories should be asked to collate current experience of the use of disease registers in their regions and to evaluate the more substantial registers. Some rationalisation and standardisation at a regional level may then be possible. Economies of scale could be achieved by amalgamating multiple registers into single regional registries. Observatories should be prepared to advise districts and PCTs on effective methods of setting up disease registers to support NSFs and other service needs. At least one, and probably several, high-level units based in academic settings should be set up to support national work on disease registers of various types. These units would be similar in conception to existing clinical trials centres. Staff would develop methods, and advise on data standards and best practice in administration, data security, and confidentiality. A generic approach would also allow data to be centrally collated to provide national information without the need to set up national registers. One centre might be set up for each of the main Saving Lives priorities. Funding source and accountability for disease registers should reflect purpose. (a) Primary care trusts, health authorities, regional specialist commissioning groups or national bodies should fund the full costs of registers the main aim of which is to improve patient care, according to the population covered. (b) Health authorities, RSCGs or relevant national bodies such as the PHLS should fund public health registers. (c) Registers for research and/or technology assessment should be set up and funded for that purpose. A new national structure for funding research registers is required. (i) A new central fund should be set up for non-capital health research infrastructure. After peer review of bids, the fund would support research registers of established national and international importance for 3- to 5-year periods. (ii) A mechanism for the assessment and support of other valuable research registers needs to be incorporated into the new Priorities and Needs funding mechanism for NHS R&D. Concerns about data protection need to be resolved urgently at a national level to allow registers to continue operating. The main action required is the drafting of suitable regulations under Section 60 of the Health and Social Care Act 2001 for discussion and consideration by the Patient Information Advisory Group and Parliament.

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Background

BACKGROUND Context 1.

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Disease registers evoke strong feelings. Epidemiologists enthuse about their potential to generate fundamental knowledge. Clinicians see them as a direct route to rational clinical practice. To public health practitioners they provide a “window” on the population. Registry staff often feel parental about “their” registers. To some patients, however, registers seem unnecessarily intrusive, an affront to their autonomy and liberty. In 1996, the NHS Executive was very interested in chronic disease management registers but “not convinced that there is a case for national … registers”.3 District population-based registers on the other hand were thought to be useful for the planning and provision of local services. Health authorities were left to decide for themselves whether they needed such registers, to fund them and to determine their form. This lack of central policy has led to confusion and inconsistency around the country. This situation now needs to be resolved. There is also a perceived need for central guidance on the role of registers in research, and clarification of responsibility for funding and regulating research registers. Current government policy firmly supports the use of disease registers to improve care and prevent ill health. In the NHS Plan, it states that “primary care trusts will identify and maintain registers of those at greatest risk from serious illness—concentrating particularly on areas where ill health is most prevalent—so that people can be offered preventive treatment” (paragraph 1.6). In the National Service Framework (NSF) for coronary heart disease (CHD) primary care teams are expected to establish and use registers of patients at risk of CHD. The diabetes NSF, due in 2001, is also likely to specify registers in some form. The particular stimulus for this report was the above statement in the White Paper Saving Lives: Our Healthier Nation on disease registers.

We will strengthen the information base on chronic diseases in the population by establishing a series of disease registers in different parts of the country. These registers will enable us to know, for example, how many people in a population are suffering from coronary heart disease, stroke, diabetes, asthma, high blood pressure. They will act as a base for the investigation into disease causes, for evaluating new ways of delivering services as well as tracking changes in disease occurrence over time. These registers will draw on the work which we have already put in train through our Information for Health programme, and will complement the existing registers for cancer. Saving Lives: Our Healthier Nation (paragraph 11.33)

Remit 5. The Department of Health commissioned this work in March 2000 with the following remit.

6 | Context

Remit


(a) Outline the potential roles of disease- or condition-based registers which would serve not only clinical but also public health ends; which would generate population-based data for incidence and prevalence analyses and aetiological studies as well as health-care evaluation. (b) Identify what is already being done at local level by working with regional contacts. (c) Identify which registers have been used and how; try to tease out criteria and conditions for a “useful” register. (d) Indicate the broad order of costs of setting up and managing a typical “useful” register at local level. (e) Indicate how “light-touch” co-ordination might take place regionally and nationally. Approach 6.

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Starting in April 2000, we undertook a selected literature review concentrating on the use of registers in diabetes, cardiovascular disease, mental health and patients receiving certain specific therapies. Cancer registries have recently been reviewed in detail by a team led by Professor Charles Gillis;4 we have not considered them in any detail. In June 2000, we wrote to all Regional Directors of Research and Development (n=8) in England, all Regional Directors of Public Health (n=8) in England and all District Directors of Public Health (n= 99) in England asking for information about disease registers currently in existence. We also asked for their views on the value of registers in public health. Other important sources included colleagues in the Department of Health, the National Research Register operated by the NHS R&D programme, Dr James Raftery of the University of Birmingham, and a draft report from the Royal College of Paediatrics and Child Health. Helpful replies were received from all the regional informants and from 61% of district directors of public health. We devised a database in which to record information about each register: fields in this database contain what we believe to be the essential information required to characterise a register, for example the case definition and the population from which the cases are drawn (see Appendix 1). For as many registers as possible, in the limited time available, we contacted a specific informant either by e-mail or telephone using a proforma of questions relating to the fields of our database. Of 234 registers now on the database, we contacted 117. Of these, 70 were able to provide virtually complete data and another 47 have provided incomplete data. Dr Sarah Garner visited four “illustrative” registers in August and September 2000 to collect more detailed information about their operation, uses and cost. In this report we present the general findings. An account of lessons from individual registers will be produced separately.

Approach | 7


Some Generic Issues in Relation to Disease Registers

SOME GENERIC ISSUES IN RELATION TO DISEASE REGISTERS Definitions and terminology Register (noun): A book in which regular entry is made of details of any kind sufficiently important to be exactly recorded. Register (verb): to set down (facts, names, etc.) formally in writing; to enter or record in a precise manner. Registry: the act of registering, registration, the place where registers are kept. Oxford English Dictionary

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Terminology in relation to disease registers and clinical databases can be confusing.5 For example, the terms register and registry are often used interchangeably. The registry is the organisation and process that supports a register and should be distinguished from the register itself. One registry may support a number of individual registers. Generic experience of running registers may be more important than specific knowledge of the topic when setting up a new register. For example, the expertise of the Danish Tuberculosis Index was used to run registers for ischaemic heart disease and stroke.5 Several specific definitions of the term register have been published in relation to disease or case registers. For example, in 1949, Bellows6 defined registers as: “a system of recording frequently used in the general field of public health, which serves as a device for the administration of programs concerned with the long-term care, follow up or observation of individual cases … (with their single distinguishing feature being) that changes in status of cases are recorded over a period of time.” More recently Brooke,6 writing for the World Health Organisation (WHO) in 1974, defined a register as: “a file of documents containing uniform information about individual persons, collected in a systematic and comprehensive way, in order to serve a predetermined purpose.” Thus, early definitions of registers emphasised a highly structured approach to data collection over a period of time and that uses for the collected information should be explicit from the outset.

Disease registers 15. In this report, we reserve the terms “disease register” and “case register” for those databases that have a clearly identified denominator population. The presence of a denominator population means that such registers can be used for epidemiological research and needs assessment, as well as to improve clinical care, and service quality and to perform technology assessment. This definition is consistent with that provided by Last in his Dictionary of Epidemiology (see below)

8 | Definitions and terminology



and is sometimes referred to as the International Epidemiological Association’s (IEA) definition. Register: In epidemiology the term register is applied to the file of data concerning all cases of a particular disease or other health-relevant condition in a defined population such that the cases can be related to a population base. … if the cases are regularly followed up, information on remission, exacerbation, prevalence, and survival can also be obtained.7 Registration: The term registration implies something more than notification for the purpose of immediate action or to permit the counting of cases. A register requires that a permanent record be established, including identifying data. Cases may be followed up, and statistical tabulations may be prepared both on frequency and on survival. In addition, the persons listed on a register may be subjects of special studies.7 16.

17.

The importance of a denominator population was also emphasised by Liam Donaldson.2 He cited four characteristics that distinguished disease registers from other collections of clinical data, namely: • registers are based on people not events; • people registered have a feature in common; • information held about these people is updated in a defined and systematic manner; • the register is based on a geographically defined population. Not all denominator populations are geographically defined, although in practice this is often the case. General practice populations make excellent denominators but have poorly defined and overlapping geographical boundaries. Occupational groups, such as doctors and nurses, can also be used, however, extrapolation from these to the general population needs to be done carefully. The IEA’s approach to the definition of registers is a development of that proposed by Pedersen for cancer registries.8 Pedersen distinguished three types of “register”: Local hospital registers—serve one hospital and are files of all patients seen at that hospital with a particular disease. Central registers—are as above, but include data from selected groups of contributing hospitals. Population-based registers—attempt to collect detailed information on all cases of a disease in a population of known size and composition. Effort is expended to identify all cases whether treated in hospital or not.

18.

What Pedersen called “local hospital registers” are still very common for diabetes but also for many other categories of patient. For example, some hospitals still maintain their own pacemaker registers. Central registers are also common. An example is the UK Cardiac Surgical Register: numbers of cases treated

Definitions and terminology | 9


19.


at each centre are collated but only national treatment rates can be calculated because catchment populations for each centre are not known. We would refer to so-called “registers” in both these categories as clinical databases (see below). Some of Pedersen’s “central registers” approximate to “population-based registers” when all centres treating the condition in a defined area are contributing data and when the condition is universally managed in hospital. A good example is the PACE project9 in which all 28 haematology consultants in the Northern NHS Region contribute information to a central registry on patients treated with haematological malignancies.

Registers that are not disease specific 20. A disease register normally identifies all cases of a particular disease in a defined population. However, some databases are set up to identify all events of a certain type (hospital admissions, GP consultations, prescriptions, deaths, etc.) in defined populations. Examples of such broadly based morbidity registers in the UK include the General Practice Research Database (GPRD formerly known as the VAMP database),10–13 the Tayside Medicines Monitoring Unit (MeMo),14,15 and the Oxford Record Linkage Study (ORLS).16–19 Similar large registers, mostly based on routine data sources of some kind, exist in Canada, the USA, Australia, New Zealand and most Scandinavian countries. 21. These non-specific registers collectively have a substantial track record of contributing to clinical and public health research. More recently, there has been much interest in using non-specific morbidity registers that include tissue samples for genetic research, for example, the Icelandic population register20 and the proposed UK Population Biomedical Collection. Nevertheless, there is a lively debate about the value of routinely collected data for research.21–23 The scope and size of routine datasets make them attractive and powerful but data quality must be adequate for the particular research purpose.24–26 22. The construction and maintenance of non-specific registers is a massive undertaking but their pluripotent nature gives them unique advantages. First, they can be used to study hypotheses that may not have been thought of at the time the database was created (for example, the cricketing example below). In these situations, answers can be produced much more quickly than from a study requiring primary data collection. There is also some protection from information bias if the data were collected and processed in ignorance of the hypothesis. There is some evidence from cross-sectional studies that left-handed people die young. The Who’s Who of Cricketers describes all “first-class” cricketers in Britain from 1864 to 1983 and lists dates of birth and death as well as the hand normally used for playing. The data in this book were used to assess longevity in 5,479 cricketers born before 1951 and for whom handedness was recorded or could be imputed—e.g. bowlers famous for bowling leg-break googlies could only be right-handed. Left-handed players died on average two years earlier than right-handed players (p=0.006). A major factor seemed to be excess risk of death for left-handed men during warfare or by accident.27 Another study using a different cricketing encyclopaedia showed no overall difference in longevity but confirmed the excess deaths due to accidents and warfare in left-handed cricketers.28



23.

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Second, the fact that the same dataset can be used for many studies across a range of fields of investigation makes them more efficient than “single-issue” registers. Finally, some of these registers (for example, the ORLS and the Icelandic database) have existed in recognisable form for decades and are an important source of data for historical trend analyses. The potential scope of a well-maintained non-specific register is enormous. The shaded box below shows the range of studies carried out using the Manitoba Health Services Commission register,24 a linked database combining health insurance claims, hospital activity, deaths and cancer registrations for the whole population. Studies carried out using the Manitoba linked data 1. Outcomes of common surgical procedures tonsillectomy hysterectomy cholecystectomy hip replacement prostatectomy coronary artery bypass graft surgery 2. Small-area variation meaning of rate variations across a number of surgical and diagnostic procedures physician supply and workload, patient utilisation 3. Physician behaviour surgical workload and turnover overall workload variations in physicians’ hospitalisation practices 4. Ageing users and non-users of health-care nursing home utilisation chiropody users subjective and objective health morbidity hypothesis”

status

and

the

“compression

of

5. Anaesthetics and surgical risk 6. Health-care utilisation in the 4 years before death 7. Effectiveness of cervical screening 26.

The Oxford Record Linkage Study contains a continuous set of linked hospital admission records and birth and death records from 1962 to 1999. These data

Definitions and terminology | 11



have been used for a comparable range of studies over the years. Notable examples include an assessment of the incidence of suicide after discharge from psychiatric hospital,29 a study of the incidence of meningitis after measles vaccination30 and a series of papers on patterns of health service utilisation, for example on geographical variations.31 Clinical databases 27. Disease or case registers are often confused with clinical databases. Table 1 summarises the main differences between these two entities. Table 1. Disease registers and clinical databases compared Disease Registers Source of cases Various and often multiple to ensure completeness

Clinical Databases Often a single institution but may be multiple, no requirement to identify all cases in any given population Denominator Defined and enumerated Not defined Follow up data Yes Yes Principal uses Patient care, aetiological studies, Patient care, Health technology needs assessment, health assessment, quality improveoutcome assessment, ment surveillance Advantages Representative distribution of Complete and accurate data on cases, can generate rates cases included “clinically-rich” Disadvantages Demanding to obtain data on Cases may be unrepresentative, all cases, difficult to obtain not possible to calculate rates consent from many cases of occurrence treated in institutions, may have to rely on routine data 28.

29.

Clinical databases do not (necessarily) relate to a defined denominator population and usually draw cases from those treated in a particular institution or group of institutions. For example, the Duke Databank for Cardiovascular Disease was established in 1969 and collected data on all patients referred to the Duke University Medical Center with suspected coronary artery disease.32 Individuals, practitioners or groups of clinicians with a common interest, are increasingly using clinical databases as part of everyday practice.33 They have been especially popular since the need for meaningful audit has been widely accepted.34,35 Clinical databases are generated when: “well-defined, discrete and continuous data elements concerning patients are routinely recorded and coupled with outcome descriptors.”32



30.


The most useful examples of clinical databases hold data on: “all consecutive cases [treated in participating clinical units] use standard definitions of conditions and outcomes, ensure data are complete and accurate, and include data on all known patient characteristics that affect outcome.”36

31.

32.

The main objectives in the use of clinical databases are quality improvement and health technology assessment. For these purposes, a defined denominator is not required. Cases on the database must be as representative as possible (individually and in terms of their distribution) of those in the target population. Databases without clear denominators cannot be used for epidemiology (i.e. to measure incidence or prevalence and to investigate causes) and are potentially misleading as indicators of the need for care in the community.37 In 2000, Cam Bowie found that about half of the consultants in one London hospital were contributing data to some form of database.33 In his small-scale pilot study, the number of databases identified (n=17) was large when compared with the number of responding consultants (n=35), giving some indication of the enormous potential for overlap and duplication in such systems. There is a need to adopt a more co-ordinated approach to the development of high-quality clinical databases as a tool for health technology assessment.21,36

Historical development of registers 33.

34.

35.

In her excellent review, Jean Weddell suggests that the Domesday Book was the first and most complete register ever made.5 It was prepared in England in 1086 to document the extent and value of royal land. Deeply unpopular at the time, the political repression implicit in Domesday was greatly resented for generations afterwards. It was not until 1753 that attempts were made to pass a Census Bill in Great Britain. This proposal for a census was described as “totally subversive to the last remains of English liberty”.5 The first Census Act was actually passed in 1800. The General Register Office was founded in 1886 “to provide means for a complete register”. Censuses have taken place every decade since but enumeration of the population still generates animosity in some quarters. A significant minority of the population resents the loss of autonomy associated with compulsory provision of information on their personal and domestic circumstances. In 1991, there were echoes of the Domesday backlash when opposition to the Community Charge (commonly referred to as the poll tax) was thought to have caused significant under-enumeration of young adult men. The registration of individuals has always been controversial. This is partly because of suspicion that the true purposes of the exercise are hidden from the public. There are historical precedents in some countries of appalling misuse of such data by totalitarian regimes. While these extremes are not expected to recur, it is important that the process of registration is overt and the public must understand and accept the justification for registration.

Historical development of registers | 13


36.

37.

38.

39.

40.


The UK census, and others like it, established mechanisms for collecting reliable denominator data. This in turn stimulated public authorities to set up systems to collect further information on particular public health problems. Perhaps the earliest example of a disease register was the National Leprosy Register set up in Norway by Royal Decree in 1856. This register was still being maintained in 1973 when it contained the names of 8,000 people although only 4 of them were still alive.38 In Britain, the registration of blind persons by Local Government Boards began in 1911. The Blind Persons Act 1920 led to a case definition and the registration of all those who qualified for grants. From 1937, all registered blind people had to be examined by an ophthalmologist. Study of the blind register revealed large numbers of elderly people blind due to cataracts providing a stimulus for the development of services to treat them. In 1968, the register was found to be 97% complete.5 A register of other disabled persons was required by the Disabled Persons (Employment) Act 1944. Registration is voluntary but the definitions used, and the rights of those registered, are laid down by statute. Cancer registration has been attempted in various countries since 1728. Effective registration was started in Massachusetts in 1927, Britain in 1930, Denmark in 1942, France in 1943, and Finland and Norway in 1952.5 In Great Britain, the Cancer Act 1939 required every local council to provide facilities for treating patients with cancer, and to keep records. The importance of population-based registries was recognised as early as 1970 in studies of cancer. Doll and colleagues in their classic monograph entitled Cancer incidence in Five Continents pointed out the need for complete ascertainment of cases, and the collection of precise contemporary data on the population at risk as well as on the cases of cancer.39 Other early registers include a psychiatric register established in 1940 in Denmark, tuberculosis registers in several countries and various longstanding registers of twins. The number of disease registers increased greatly in the 1950s for a variety of reasons. There was increasing concern about the impact of chronic diseases, such as diabetes and CHD. Epidemiologists were developing new methods of analysis that went beyond those used for infectious diseases.6 Furthermore, advances in information technology made the handling of large amounts of data much easier and less expensive.

Current uses of registers Patient Care 41. Databases and registers are widely used by individual practitioners and groups of professionals to improve the quality of clinical care that they provide to patients. Registers are used to target resources and ensure consistency and complete coverage. The particular ways in which registers are used are listed in the following paragraphs. 42. Databases used for managing patients can be relatively undemanding in terms of information needs. A loose case definition may be adequate; there is no need to relate the cases to a specific denominator population. There is also no need actively to seek cases that are not already apparent through normal clinical channels. Data protection issues are important for these databases although it

14 | Historical development of registers



will normally be relatively easy to obtain appropriate consent (implied or explicit). It is also particularly important that patient contact details do not become obsolete. These registers or databases become rather more complicated to administer when they are shared by a group of clinicians. In such instances, it is important to specify standard definitions and practices in order to maintain comparability. However, the potential to use shared databases for comparative audit may well justify the extra work involved. Regular review and recall 43. The most obvious use of a register is as an aide to the management of patients who have a continuing need for health care. Clinicians find it helpful to compile lists of patients who need regular review. Regular review may be necessary because they receive longterm therapy (e.g. patients with epilepsy, leprosy, diabetes, hydrocephalus or acne), because they need to be screened for treatable complications of their disease (e.g. patients with diabetes, renal failure, inflammatory bowel disease, lichen sclerosus, polyposis coli or melanoma) or to encourage and educate patients to adopt a health-promoting lifestyle (e.g. patients at risk of CHD or who have a mental health problem). Structured care programmes (including evaluation) 44. If the aim is to provide structured and comprehensive care to a population, then a disease register is probably an essential prerequisite and definitely required to know whether that aim has been achieved. The most common example of registers being used in this way is in the care of patients with diabetes. Similar registers are currently being set up for the care of patients with CHD in response to the NSF. These will be used, for example, to ensure that all patients discharged from hospital after a myocardial infarction receive appropriate secondary prevention advice and treatment. Monitoring high risk groups 45. Registers can be set up to monitor patients who are at high risk of complications due to some specific exposure (e.g. patients who have had thyroid treatment, have been treated with human growth hormone, or patients who have received radiotherapy). Also, children born very prematurely are monitored closely for signs of developmental disorder because there is evidence that early intervention is effective. Social services registers of children “at risk” from abuse are a comparable example. 46. There is an increasing range of devices that can be implanted in various parts of the body, from cochlear implants to automatic defibrillators. As notices are occasionally issued in relation to these devices, it is essential to have an up-to-date register of relevant patients. This need came to light when the Bjork-Shiley heart valve was found to be faulty. More recently breast implants have been a cause for concern. It is, however, hard to maintain a device register when much of the activity takes place in the private sector and patients are extremely sensitive about confidentiality, as is the case for breast implants.

Current uses of registers | 15



Managing demand and regulating access 47. Another function of registers in clinical practice is as a means of regulating access to services according to specified criteria. For example, patients who are registered blind or disabled are entitled to certain benefits as a result. Communication 48. Registers can be used to improve clinical care by acting as a focus for a community-wide collaborative approach to the management of particular groups of patients. The existence of a register leads to the sharing of information about practice, and the standardisation of practice around explicit clinical guidelines or a consensus standard. The register also encourages audit and facilitates research. An example of this approach is the early use of a diabetic register in Poole to encourage shared care between hospital and primary care.40 Another example is the register developed for haematological malignancies as part of the PACE project in the Northern Region—held up as a generic model for this type of approach.9 Risk stratification 49. Information from registers has been used to improve the diagnostic process and can be incorporated into systems that provide a computer-assisted diagnosis. Registers, such as the Framingham study, also provide the basic data used to construct risk prediction nomograms or formulas. This and other prognostic information derived from registers can be shared with patients allowing them to be involved in decisions about their own management. For example, if patients are to give informed consent to a surgical procedure it is essential that they know how common various outcomes are in individuals similar to them. This information is rarely available from clinical trials, which generally do not contain enough data to give precise estimates of outcomes in subgroups. Large-scale person-based registers can often provide that information. However, caution needs to be exercised when extrapolating from register results to individual patients. In general, prognostic models based on observational data should be seen as illustrative rather than predictive.41 Public Health 50. Registers are important public health tools. Access to reliable population-based data on incidence, prevalence and outcome is essential to public health practice. Eva Alberman has described four distinct public health functions for disease registers.42 All of these require registers with defined denominator populations and complete or at least unbiased identification of cases. For example, in the case of congenital anomaly registers it may be necessary to obtain data on terminations and prenatal deaths for both the numerator and denominator (i.e. affected and unaffected conceptions). Surveillance of health and disease 51. Altered patterns of registration over time, place or between population groups may suggest new causes of disease or document the effects of old ones. For example, national monitoring of cases of Reye’s syndrome by the British

16 | Current uses of registers


52.

53.


Paediatric Surveillance Unit, and the Communicable Disease Surveillance Centre, provided evidence that aspirin may be the cause of many cases of the syndrome.43–45 The occurrence of clusters of cancers, infectious diseases or congenital anomalies may identify a point source responsible for an outbreak and lead directly to preventive action. If an association is suspected, the register can act as an unbiased sampling frame from which to select cases for a case-control study. Non-specific registers may also be able to supply large numbers of suitable controls. Negative findings are also important for reassurance. For example, there was some concern that hip replacement might increase the risk of lymphoma because some tumours were found to contain fragments of metal from hip prostheses. Record linkage studies using cancer and hospital admission registers showed no association.46 One reason it is difficult to refute the suggested relationship between MMR vaccine and inflammatory bowel disease or autism is that adequate registers of these two diseases did not exist prior to almost universal exposure to MMR vaccine.47

Planning the provision of health care 54. The diseases for which the government is developing NSFs are associated with approximately 50% of all health-care expenditure. An appropriate system of population-based disease registers for diabetes, CHD, stroke, cancer and major mental health problems would provide the essential intelligence required for proper capital planning, workforce planning and general service configuration. For example, the demand for cardiac catheterisation and revascularisation in a given population over the next decade could be predicted with reasonable accuracy from an adequate CHD register. 55. Such registers do not need to be comprehensive in terms of national population coverage, because errors of extrapolation are likely to be small compared with other uncertainties. More important is that estimates derived from the chosen sample populations are accurate (free from measurement error) and precise (based on adequate numbers of individuals). For “common diseases” therefore, the priority is that registers are of consistently high quality and completeness. Also the distribution of the registers should be representative of the national population. 56. For some other conditions, individual patients can make high demands on local health services. For this reason, it is useful to monitor trends in severely disabling conditions such as serious head injuries, progressive neurological conditions and some congenital anomalies. It is also important to predict the demand for expensive high-technology interventions, such as renal replacement therapy or bone marrow transplantation associated with identifiable clinical groups. Monitoring the burden of ill health in the population 57. From the public health perspective, it is important to know the relative burden of morbidity attributable to different diseases in the population. This information is crucial in guiding policy on resource allocation for treatment services and for prevention and research. 58. Registers can also be used to assess outcomes as part of an audit, for example




in comparing survival in cancer patients treated in different units. They can also provide data on relative rates of survival or morbidity after different procedures. Monitoring the impact of preventive measures 59. Registers are particularly good for monitoring the outcome of interventions aimed at the whole population such as screening programmes for breast cancer, cervical cancer, inherited metabolic diseases, and congenital anomalies. Registers can also be used to monitor the effectiveness of other preventive campaigns. For example, registering all cases of Creutzfeld-Jakob disease is an important part of managing the consequences of the bovine spongiform encephalitis epidemic. Diabetes registers can be used to audit the uptake and effectiveness of preventive care for diabetics. Infectious disease notifications can be used as an indicator of the effectiveness of public health measures to prevent sexually transmitted diseases and imported tropical diseases such as malaria. Congenital anomaly registers provide a mechanism by which it is possible to monitor the success of campaigns to encourage periconceptual folic acid supplements, and the uptake of rubella vaccination programmes. Technology Assessment 60. Medical technology has advanced more rapidly than the capacity to evaluate it. Randomised controlled trials may take too long and be too specific to answer all the questions raised by new technologies. They are also expensive. Databases and registers can make a useful contribution to this unmet need for evaluation if they conform to criteria of the sort put forward by Pryor et al (see below). “To be most useful for technology evaluations … a data bank requires a large number of patients, defined identically, about whom data are uniformly collected. For chronic conditions, patients must be followed over time using precise definitions applied uniformly by different investigators and over long periods of time.” Pryor et al, 198532 61.

62.

Technology assessments using databases and registers are not as rigorous as randomised trials but can complement more selective randomised studies. They have the advantage of being based on more representative populations than most randomised studies and often include far larger numbers of cases. For example, most of the reliable information about prognosis after coronary artery bypass surgery comes from analysis of clinical databases and registers not randomised trials. The BCIS Interventional Cardiology Database has shown that use of stents during angioplasty reduces the need for emergency bypass surgery. The Medical Devices Agency has funded several device registers on the basis that they provide valuable data on the relative merits of different designs in different patient groups. Technologies that have been evaluated using registers include drugs, devices and diagnostic tests. The GPRD has been used extensively to assess the incidence of unusual side effects of drugs, for example, the incidence of venous

18 | Current uses of registers



thrombo-embolism in users of oral contraceptives, and the incidence of severe skin reactions in patients taking sulphonamide antibiotics. Research 63. Pryor et al32 distinguish four areas in which registers contribute to research (in addition to technology assessment). (a) Descriptive studies—registers provide accurate numbers of patients receiving certain treatments, diagnostic tests or experiencing defined outcomes in representative groups of patients. If the register is truly population-based and complete in its coverage, these data can be expressed as population incidence and prevalence rates. For example the Duke database shows that the mortality of patients referred for cardiac catheterisation has declined over time. This is an interesting and “true” finding despite the fact that the database does not necessarily show why mortality has declined. (b) Improving the performance of other research designs such as clinical trials and epidemiological studies—registers can be used to develop trial protocols and assist in the conduct of trials and other studies in any one of the following ways: (i) the development of methods of known reliability for making observations; (ii) improved compliance with research protocols between sites (i.e. provision of information on cases not entered in trials); (iii) identification of subgroups of patients for a particular therapy, cases for a case-control study or cohorts with a given exposure criterion; (iv) identification of suitable controls, matched for appropriate confounders; (v) provision of data to calculate sample sizes; (vi) identification and quantification of the effect of potential confounders that may be associated with outcomes of interest; (vii) investigation of interaction between variables of interest; (viii) development and testing of analytical techniques for application in other settings. (c) Studies of processes – registers provide data that can be used to test agreement between observers or the performance of diagnostic tests of various kinds. (d) Hypothesis testing—registers can be used to test hypotheses but the problem with this research strategy is that apparent therapeutic effects may be due to unrecognised factors affecting treatment allocation. For example register studies show an apparent reduction in rates of CHD in women who take postmenopausal hormone replacement therapy, which has not been reproduced in randomised trials.48 Thus, the use of registers to test hypotheses in non-randomised studies is best restricted to those situations in which randomised studies are either not possible or unlikely ever to be performed.49 64. Register-based non-randomised studies may add important information to the results of randomised studies. Registers usually cover a wider population, are larger and provide more prolonged follow-up. Whatever the arguments about the relative merits of randomised and non-randomised studies, some hypothe-




ses can only be tested in longstanding prospective non-specific databases. For example, a recent investigation using the Oxford Record Linkage Study considered the risk of breast cancer in relation to past experience of termination of pregnancy.50 The characteristics of successful registers 65.

Some registers are much more successful than others. Many, despite worthwhile objectives, fail to realise their potential. However, some failures are predictable. Certain factors seem consistently to predict success.

Multidisciplinary 66. The skills required to design and manage a successful register will rarely be found in one individual. The team should normally include a computer scientist, biostatistician, and someone trained in the clinical domain of the register. The best teams are said to include a number of people whose skills cross traditional occupational boundaries – for example, a statistically or IT-trained clinician.

Pryor et al described factors that predict success for a register as follows:32 • • • • •

appropriate multidisciplinary team; stable funding; focused aims; data collection systems and design that relate well to function; relevant leadership.

Stable funding 67. Funding is important. The amount of money required to set up and maintain a register is not trivial (see below), and it may be years before the benefits of the register are apparent. Much work can be wasted if an interruption to funding leads to loss of key staff or breaks in the data collection process. Stability of funding does not necessarily imply one source: it may be achieved by progressively replacing one form of funding with another, for example as the project matures from a pilot to a substantive register. Focused aims 68. A clear focus on the initial aims of the register is crucial. There is a temptation to collect large amounts of data that may be of marginal value and questionable quality. This is often a mistake and can interfere with the primary objectives of the register.

20 | The characteristics of successful registers



“The temptation to collect information is hard to resist.” “The most successful registers seem to be those in which the data collected are accurate, restricted to the essentials, and meet a need that cannot be satisfied in any other way. …information must be collected from as many sources as possible, and checked for completeness and duplication.” Weddell, 19735

Methods related to function 69. Ultimately, the value of the database should be apparent to someone whose needs it was designed to meet. The register must be prepared to change its methods in order to be responsive to changes in the needs of its users. Leadership 70. There should be a clearly identified senior professional in charge of the registry. This person may be a clinical specialist, epidemiologist or statistician. There should also be a senior administrative officer who is responsible for the quality of all aspects of the register. Other criteria 71. Criteria for an ideal physical location for a research register have been prepared by a working party of the Royal College of Paediatrics and Child Health.51

Criteria for an ideal physical location for a research register:51 • An academic environment • Statistical and epidemiological expertise available • “Neutral ground” protected from possible rivalries • a place where all potential users feel they have equal access • a place where all disciplines feel comfortable • a place where all those with legitimate scientific interests feel on an equal footing

72.

In the context of a general practice diabetes register, Cheales and Howitt1 suggested some key factors necessary for success as follows. (a) Clear and tangible benefits must be demonstrated from the outset for the patients involved and the practices delivering their care. For example, a register might help with call and recall, highlight defaulters and help co-ordinate care between agencies. (b) The precise functions of the register should reflect local variations in the arrangements for providing care.

The characteristics of successful registers | 21



(c) All disciplines involved in providing care (e.g. practice nurses, chiropodists, and doctors) should be involved and understand the register. (d) Data collected should be kept to a minimum and duplication of data entry avoided if at all possible. (This implies that the register will be kept compatible with current general practice computing systems.) (e) Concise and relevant information should be fed back to the practices. There should be scope to answer ad hoc queries from practices. (f) Educational support should be provided to the practices. (g) Practices may need reimbursement for the work involved in providing data, and resources may be required to meet the additional need for care identified as a result of using the register. Both types of resource requirement should be considered in advance of the establishment of the register. (h) Issues of confidentiality, data protection and consent must be addressed in detail from the beginning (see below). In particular, any concern that practice performance might be policed via the register must be dispelled.

How to set up a disease register

“To set up a district diabetes register on a routine and long-term basis across an entire district is a formidable undertaking that should not be underestimated.” Vaughan, 199652

73.

The following notes refer to the steps that might be considered when setting up a population-based register. They draw on the report from the Royal College of Paediatrics51 as well as other published work.

Set up an Expert Group 74. The Expert Group will guide the future development of the registry and ensure that the register(s) have a sound financial and scientific basis. The Expert Group should decide on initial data collection processes and data content. The Expert Group should be convened for a defined period of time as many of its functions will be taken over by the Steering Group when the register is fully established. Set up a Steering Group 75. The Steering Group should ensure that the register is run according to its stated aims and objectives, and that the rights of patients are respected. The Steering Group should oversee the use of register data, and approve any additional research projects. Patients and their families should be represented on this group, as should relevant clinical, research and administrative interests. The Chair should be someone regarded as “neutral” to any professional divisions or other vested interests.

22 | How to set up a disease register



Notify the Information Commissioner 76. Under the Data Protection Act 1998, starting a register is a form of “processing” of personal data and must be notified to the Information Commissioner (used to be known as the Data Protection Commissioner). The Data Controller, defined as the person who determines the purposes for which, and the manner in which, any data are processed, should make the notification. The Commissioner will require a description of the data to be collected and the purposes to which the data will be put. Obtain approval from a Research Ethics Committee 77. If the register is to be used for research, an appropriate Research Ethics Committee should approve its establishment. Any specific projects not referred to in the original application will need separate approval. If the register is solely used for research purposes, it may be exempt from some of the requirements of the Data Protection Act 1998 (for example, the obligation to provide copies of any data held on them to individuals who might request it). Establish arrangements for access to the data 78. Thought should be given to the likely spectrum of users of the data. It may be that certain specialist groups, or an in-house group, are going to be the major users. However, it is important to ensure equitable access for those who make best use of the data with respect to the original purposes of the register. It may be useful to set up a “user group” who would share experience and expertise in the use of the data and would work closely with the registry itself. The user group should include the main clinical and research groups active in the specialist area covered by the register. Consider arrangements for data security 79. The register should be in a secure room within a generally secure building. Apart from physical security, other types of security include logical security (encryption), technical security (passwords) and procedural security (staff training and written records of procedures). Establish appropriate arrangements for accountability 80. Officers responsible for the registry should report to the institution that houses it and the organisation that funds it. The Data Controller is also legally accountable to the Information Commissioner. Annual reports and regular feedback to those contributing data are essential to maintain interest. Publicise the registry and its findings 81. It is essential to promote the register among those who contribute data. Some form of “corporate identity” may be useful to reinforce the presence of the register. Maintaining goodwill also requires frequent communication with participants and appropriate reporting whether at scientific meetings or through professional publications. It is essential to avoid the impression that the data “just sit there!”.

How to set up a disease register | 23



Some data considerations 82. A clear case definition is fundamental to the success of any register. The case definition must include guidance on which cases should be included and which excluded, including cut-off points for any test or other indicator of severity. A protocol for how to deal with multiple diseases also needs to be established. For example, the same individual may have more than one congenital anomaly or cancer. Diseases for which a clear case definition is hard to implement reliably are not suitable subjects for disease registers. 83. The methods used to ascertain cases need to be considered carefully. It is important to grasp that the nature of the cases on a register will reflect the method chosen to ascertain them. Whichever method is chosen, the diagnostic process must be based on a formal protocol. Although laboratory results provide a robust method of identifying cases, for some diseases no definitive test exists and clinical assessment is the basis for a diagnosis. In this case, it is good practice to assess the reliability of the diagnostic protocol from the outset. 84. Full ascertainment can rarely be achieved from a single source. Thus, for diabetes registers, data must be obtained from both primary and secondary care to ensure that all cases in a population have been identified (see quote below). In fact, it is good practice to have as many independent sources of ascertainment as possible. This approach is designed to achieve over-ascertainment—duplicate notifications must then be identified and deleted.

“For many conditions, including diabetes, any population-based register will need to include information obtained from general practice if it hopes to be comprehensive in its coverage.” Cheales and Howitt, 19961

85.

86.

The reliable identification of duplicates requires patient identifiers otherwise the same case may be registered twice. However, the identification of duplicates is not straightforward even when patient identifiers are available. For example, people may change their surname and address at the same time when they marry. Twins may have identical or very similar dates of birth, addresses and names. For names that are common, sooner or later two cases will have the same name and birth date—has one person moved or are they two different people? There is a considerable legacy of theory and practice in record linkage that can be used to help resolve these issues within well-defined levels of uncertainty.53 The introduction of the NHS number will help but it is not yet sufficiently widely used to obviate the need for probabilistic record linkage techniques. The future development of disease registers should relate to the evolving NHS information strategy. The strategy has recently been updated in the document Building the Information Core (www.doh.gov.uk/ipu/strategy/update/index.htm). With the introduction of electronic patient records, relevant clinical information will be more comprehensively and consistently available in an electronic form. Electronic health records will be even more helpful to registers as data from a number of sources will be brought together in a linked person-based record. The adoption of NHS-wide information standards should also lead to more consis-




tent levels of ascertainment and specificity for registers based on routine data sources. It is important that registers take advantage of these developments in their planning and design. They should not, for example, produce their own data standards when national ones would do just as well. Obtaining proper consent 87. Wherever possible, explicit consent must be obtained from those whose information is to be held on registers (or from their parents or other guardians). The person obtaining consent should be the clinician caring for the patient. Relationships between patients and the registry are likely to be much stronger if explicit consent has been obtained. 88. A third party compiling a register that might influence the clinical care of the patients on it must obtain explicit consent from those individuals. This requirement also applies to any register for which contact with patients is necessary for data collection, for example, to obtain follow-up data. For registers where patients will not be contacted, and where the purpose is limited to research, a form of implicit consent may be sufficient but only if explicit consent could not be obtained for some reason. 89. The requirement to inform patients about a register can also be relaxed if providing such information would require disproportionate effort. For example, a large register that relies on secondary data sources would not be able to seek consent from all the individuals concerned. Indeed, this process could introduce bias because of systematic differences in the ability to contact some groups of individuals (e.g. the unemployed or students). 90. The statutory framework in this area is complex and evolving. Many of the specific requirements of the Data Protection Act 1998 are open to interpretation (for example, the meaning of “disproportionate effort”), and there is an absence of relevant case law. The GMC has published an interpretation, that has led many doctors to believe they risk legal action and/or GMC action against them if they disclose information to registries without the explicit consent of patients (see section below on data protection and confidentiality). The MRC has also published guidance on the legal position.54 91. Most identifiable health data would be defined by the Data Protection Act 1998 as “sensitive” personal data. In Schedule 3 of the Act, conditions under which processing or further processing of sensitive personal data (which would include compiling a register) can take place are set out. There are two conditions that are likely to be satisfied in the case of disease registers and which could be used to justify the process: (a) either the data subject has given their explicit consent; or (b) processing is necessary for medical purposes (including preventive medicine, medical diagnosis, medical research, the provision of care and treatment and the management of health-care services) and is undertaken by: (i) a health professional (defined by the Act); or (ii) a person who owes an equivalent duty of confidentiality. 92. For NHS bodies, there should be no difficulty in satisfying the second condition—namely, that processing data is necessary—and therefore consent would not be needed for normal data use. Likewise, a GP who set up a diabetes register in his or her own practice would be on relatively safe ground. It would be

How to set up a disease register | 25


93.

94.

95.

96.

97.


more difficult to justify the establishment of a register compiled by a third party from records supplied by a range of clinicians (e.g. a cancer registry or other central database). To avoid any possibility of legal challenge about what is “necessary for medical purposes” it would be wise to obtain consent for any use of personal data. However, methodological problems quickly arise if consent rates fall below 100%: it is probable that non-compliant individuals will differ from those who are compliant in a systematic way.55 What is the precise meaning of “consent” under the Data Protection Act 1998? In the Act “Consent” is not defined but it is in the European Directive that gave rise to it, as follows: [a subject’s consent is] “any freely given specific and informed indication of his wishes by which the data subject signifies his agreement to personal data relating to him being processed.” The use of the term “signify” implies some form of active communication between the parties. Thus, data controllers cannot infer consent from nonresponse to a communication. Moreover consent obtained on the basis of misleading information would not be valid. Consent may subsequently be withdrawn. The requirement for “explicit” consent for processing of sensitive data (such as ethnic group or medical diagnoses) suggests that the consent of the subject must be absolutely clear. In appropriate cases, the consent should cover the type of data to be processed, and the purposes of any processing and disclosures that may be made. The Department of Health has three definitions of consent: (a) Consent—Agreement, either express or implied, to an action based on knowledge of what the action involves, its likely consequences and the option of saying no. (b) Express Consent—Consent which is expressed orally or in writing (except where patients cannot write or speak, when other forms of communication may be sufficient) (GMC). (c) Implied Consent—Consent which is inferred from a person’s conduct in the light of facts and matters which they are aware of, or ought reasonably to be aware of, including the option of saying no. Difficulties arise because the form of consent required will vary depending on the situation and the relative “sensitivity” of the data. In some cases, implied consent may be sufficient but this has not been tested in the courts. The greater the level of ambiguity about the form of consent, the greater the likelihood that its validity could be challenged. Implied consent may be attractive but it carries a significant risk of legal challenge. The validity of any form of consent would need to be assessed in the light of individual circumstances. An important element in obtaining consent is the provision of appropriate information prior to consent being obtained, and normally some time before consent is sought. Information given to patients before they consent should contain as much detail as possible on the following (adapted from Thornes):51 (a) The purposes of the register and of any data processing involved. (b) What data will be held. (c) How further data will be obtained (e.g. by linkage with other datasets). (d) How the data will be kept secure.



98.


(e) Contact details for the data controller (name, telephone number, address) should they wish to check their details on the register. (f) Confirmation that individuals will not be identified in any publications. (g) How they will be informed about findings from the register. (h) Details about how contacts will be made (usually only through known clinicians but it may sometimes be appropriate for the register to contact individuals directly). No pressure should be exerted to obtain consent. It must be made clear that treatment will not be prejudiced if patients decide against registration, although the benefits of registration should be explained. Consent or refusal should be noted in the medical record and should be respected unless overridden by the law.

Evaluating the quality and value of registers 99.

Registers should be set up only to study important health problems, and the systems used should achieve their purpose efficiently.56 Registers are expensive to set up and maintain in terms of money and time. They also have substantial opportunity costs for professionals and for patients. They may require some loss of patient autonomy in the use of personal data. For all these reasons, it is essential that registers are established with clearly stated worthwhile objectives, and that the objectives are capable of being realised by the registry system as it operates in practice, as outlined by Weddell:

“The value of a register must be examined at intervals to ensure that the objectives still hold and are being met. If they are not, the objectives should be revised or the register closed.” (Weddell, 1973)

100. At a strategic level, it is important to allocate resources for registers in a balanced way. Funded registers should each be fulfilling a useful public health function, and duplication of effort should be avoided by co-ordinating the work of individual registries. It is also important that appropriate methods are used according to the issue being studied and the key purpose(s) of the register. Finally, the usefulness of registers is dependent on the quality of their design and of the data that they contain. Any public health system that relies on disease registers must ensure that the individual component registers are of adequate quality.6,56 Quality assessment 101. Detailed guidance on how to assess the quality of a surveillance system is given in an article by Klaucke and his colleagues from the US Centre for Disease Control.56 This approach has been widely accepted as a standard, especially in the US, and can be used for disease registers with slight adaptation.

Evaluating the quality and value of registers | 27



The process of evaluation of a register should include the following56 • an assessment of the public health importance of the topic • a description of the systems used and their objectives; • a description of the uses and other outputs of the register; • an evaluation of the system against each of the attributes given below; • a description of the resources used to operate the system; • a summary of whether the system is meeting its objectives and any modifications required

102. An abbreviated approach to evaluation is described by Goldberg et al.6 The evaluation is divided into an assessment of a register’s completeness and of its validity. Completeness is defined as the proportion of all cases in the population that appear in the register. If completeness is less than 100% (the usual situation) it is also important to assess any systematic biases in the level of reporting. Validity in this context is summarised as the extent to which the information on the register agrees with some external source which objectively measures the same variable.6 103. Three methods are discussed by Golderg et al for assessing completeness. (a) Death certificate method, in which completeness is the proportion of cases not first identified from a death certificate. If cases are identified only at death, it means that throughout life they must have eluded the normal registration processes. This approach has been used a lot by cancer registries but also by stroke and CHD registers. (b) Independent case ascertainment, in which completeness is assessed by means of an independent survey, has also been used widely. A survey of part of the population covered by the register can be used to impute an approximate measure of completeness for the whole register. Another related approach is to use capture-recapture techniques to compare two incomplete registers.57 For this method to be valid, the two registers must be assumed to have identified independent random samples of the population of true cases. Unfortunately, this rarely holds in practice. (c) Historic data method, in which data from previous years, or some other “known” prevalence or incidence rate, are used to predict an expected rate of identification. This approach is useful for general practice diabetes registers when the expected prevalence is taken to be at least 2%.

28 | Evaluating the quality and value of registers



The Attributes of Disease Registers (adapted from Klaucke et al56 and Rothman58) Sensitivity: To what extent does the system identify all of the events in the target population? For purposes of monitoring trends, low sensitivity may be acceptable if sensitivity is consistent over time and detected events are representative. For purposes of assessing the impact of a health problem, high sensitivity is required. Timeliness: This attribute refers to the entire cycle of information flow, ranging from information collection to dissemination. The need for timeliness depends on the public health urgency of a problem and the types of interventions that are available. Representativeness: To what extent do events detected through the surveillance system represent persons with the condition of interest in the target population? A lack of representativeness may lead to misallocation of health resources. Predictive Value: To what extent are reported cases really cases? To what extent do measured changes in trends reflect of events in the community? Accuracy and Completeness of Descriptive Information: Forms for reporting health events often include descriptive personal information, such as demographic characteristics, clinical pattern of disease, or potential exposures. To what extent are these sections of forms completed? Is the information sufficiently reliable? Simplicity: Are forms easy to complete? Are procedures unobtrusive? Is software “user-friendly”? Is data collection kept to a necessary minimum? Flexibility: Can the system change to address new questions? Can it adapt to evolving standards of diagnosis or medical care? Acceptability: To what extent are the participants in a surveillance system (those who report cases, welcome staff into their hospitals or offices, complete forms, etc.) enthusiastic about the system? Does the effort they invest yield useful information? 104. Goldberg et al also described three broad methods for assessing validity.6 (a) Diagnostic criteria method, in which registry staff examine clinical records to assess the proportion of cases that satisfy strict diagnostic criteria. For example, what proportion of cases of myocardial infarction have elevated enzymes or what proportion of patients with stroke have had a confirmatory CT or MRI scan. (b) Re-abstracted record method, in which for a sample of cases data are reabstracted from the original source records and compared with register records. Re-abstraction is done under controlled circumstances, and is assumed to be correct. (c) Internal consistency method, in which a computer programme is used to check the register for illegitimate codes and logical inconsistencies in the data.

Evaluating the quality and value of registers | 29


Confidentiality and Data Protection

CONFIDENTIALITY AND DATA PROTECTION 105. In a thoughtful essay that predates the current debate, Roger Higgs discusses the ethical background to the use of personal health data in disease registers.59 He argues that the basis for our ideas about confidentiality is as follows.

Human beings have a right to privacy in a humane society but this right is not absolute in either an ethical or a legal sense. Adapted from Higgs, 1996

106. Higgs explores the balance of rights between individuals and society and acknowledges that this presents some intrinsic problems for the operation of registers.59 It is also important to reconcile the principle of confidentiality, as expressed in the Hippocratic oath below, with modern multidisciplinary clinical practice in which sensitive information is routinely shared among several professionals.

“All that comes to my knowledge in the exercise of my profession, or outside of my profession, or in my daily commerce with people, which ought not to be spread abroad, I will keep secret and never reveal.” The Hippocratic Oath

107. Patients’ fears about confidentiality need to be respected and addressed. Once information has been transferred to a registry, it is outwith the individual’s control and beyond the control of anyone who knows them. This is a cause for concern because: (a) the information may be taken out of context and thereby misinterpreted; (b) the information may be wrong, and mistakes may not be detected; (c) errors once identified can be difficult to eradicate and may remain in the system e.g. on back up copies; (d) registers are hard to police and therefore data controllers must operate on trust; (e) it may not be clear who retains responsibility for the common law duty of confidentiality to patients. 108. Thus, there are legitimate reasons to control and restrict the use of sensitive personal data to those purposes that are both necessary and important. However, the loss of individual autonomy and small increase in the risk of a breach of privacy, associated with any transfer of sensitive data, sometimes have to be accepted as the “cost” of undertaking an important function such as the surveillance of infectious diseases or the investigation of drug safety. Difficulties arise because these costs fall on individuals whereas the benefits accrue to society as a whole.

30 | Confidentiality and Data Protection



107. Patients’ fears about confidentiality need to be respected and addressed. Once information has been transferred to a registry, it is outwith the individual’s control and beyond the control of anyone who knows them. This is a cause for concern because: (a) the information may be taken out of context and thereby misinterpreted; (b) the information may be wrong, and mistakes may not be detected; (c) errors once identified can be difficult to eradicate and may remain in the system e.g. on back up copies; (d) registers are hard to police and therefore data controllers must operate on trust; (e) it may not be clear who retains responsibility for the common law duty of confidentiality to patients. 108. Thus, there are legitimate reasons to control and restrict the use of sensitive personal data to those purposes that are both necessary and important. However, the loss of individual autonomy and small increase in the risk of a breach of privacy, associated with any transfer of sensitive data, sometimes have to be accepted as the “cost” of undertaking an important function such as the surveillance of infectious diseases or the investigation of drug safety. Difficulties arise because these costs fall on individuals whereas the benefits accrue to society as a whole. Medical Research Council Guidance 109. In 1985, the Medical Research Council set out its view on the use of confidential information for research.60 Crucially, the MRC stated that, provided every practicable step had been taken to safeguard confidentiality and to ensure that no disadvantage, harm, distress, or embarrassment is suffered by any individual as a consequence, there should be no impediment to the use of personal medical information in research. The justification for this view was that the potential and actual value of research was great enough to override any loss of autonomy associated with the use of such information for research purposes. 110. Moreover, the MRC pointed out that the confidential transfer of medical information is a necessary and accepted part of normal practice in healthcare and medical research. The transfer of data with appropriate safeguards to another doctor for research purposes was not seen as contrary to the duty of confidentiality owed by all health professionals to their patients. The MRC considered that, subject to scrupulous safeguards about confidentiality, information about patients could be made available for medical research without their explicit consent. These guidelines form the basis on which many current registers have been set up. 111. The MRC updated its guidance in 2000, partly to take account of the Data Protection Act 1998.54 This guidance is now slightly out of date because of very recent legislation although it remains helpful and authoritative. It covers the main issues facing researchers who wish to use personal information, namely: (a) The collection of information from subjects in trials or other patient-based research;

Medical Research Council Guidance | 31



(b) the use of data from routine health records to identify potential subjects for patient-based research; (c) the analysis of patterns of health and disease using existing health records (whether in paper or electronic format); (d) the study of people who have a particular job or lifestyle. 112. The MRC defines three categories of data normally used in research which are of differing sensitivity. (a) Coded information—this category of data contains information that could readily be used to identify people but is concealed by coding. The research team holds the key to the code. (b) Linked anonymised data—this category of data is anonymous to the research team but contains coded data that could be used to identify people (e.g. the NHS number). Others outside the research team (e.g. the clinical carer) hold the key to this code. (c) Unlinked anonymised data—this category of data contains nothing that could reasonably be used to identify people: the link to individuals has been irreversibly broken. 113. Registers should use anonymised data wherever possible, although it is hard to see how a register could fulfil its function using unlinked anonymised data. We identified several examples of registers in which linked anonymised data are being used satisfactorily. However, this approach places an additional administrative burden and responsibility for data quality on the agency supplying the data. In paragraph 5.1.6 of the MRC guidance, there is useful advice on this issue (see below). The MRC points out that removal of obvious identifiers such as names and addresses does not guarantee anonymity. For this reason, ethical use and data security considerations remain important even for anonymous datasets

“If it is practical and reliable, the removal, or coding, of identifying information should be done within the team or organisation responsible for the individual’s care. Where this is not possible, it is preferable for a member of the research team to help with the anonymisation rather than for identifiable information to be used.” Paragraph 5.1.6, MRC, 200054

114. MRC guidance on use of data disclosed without consent is particularly relevant to some registers. In cases where it is impracticable to obtain consent for disclosure, there are three principles on which further guidance is based. (a) Hospitals and general practices must make patients aware in general terms that their data may be used for research and under what circumstances. Any objections should be respected. (b) Data can be disclosed without consent only when:* (i) the likely benefits to society outweigh the implications of the loss of confidentiality [by a clear margin];

32 | Medical Research Council



(ii) there is no intention to feed information back to individuals or take decisions that affect them; and (iii) there are no practicable alternatives [i.e. completely different study designs] of equal effectiveness. (c) The infringement of confidentiality should be kept to a minimum. 115. It should be noted that compliance with MRC guidance does not protect registers from legal challenge. It is also essential to understand the implications of the Data Protection Act 1998, the Human Rights Act 1998 and Common Law with respect to any proposed use of personal information obtained in confidence. However, the legal position is currently unsatisfactory in that there are contradictions and uncertainties within the legislative framework. This situation will be resolved in the near future (see below) but in the meantime the MRC guidance seems a reasonable basis for practice.

The Data Protection Act 1998 116. The main provisions of the Data Protection Act 1998 came into force in March 2000. They are broader than the equivalent provisions under the Data Protection Act 1984 and more stringent with respect to the conditions placed on Data Controllers. The Data Protection Act 1998 gives effect to EC Directive 95/46/EC. 117. Under the Data Protection Act 1998 almost all data held by registries would be defined as “sensitive personal data” and almost any use of the data would be defined as “processing” or “further processing”. Importantly, in the Act, the disclosure of information is included under the compendious term “processing”. This has led to some confusion as follows: “It seems that I can process the data but I cannot disclose it.” The registry itself is likely to be the “Data Controller.”. 118. The main principles of the Act are given below. Satisfying the first “fair processing” principle is the main challenge for disease registers. As part of “fair processing”, information should be provided to data subjects about the use of the data. Data controllers are not exempt from this condition simply because they did not obtain the data direct from the subjects—for example, if the data came from routine information systems. However, they are not required to provide this information if to do so would require “disproportionate” effort. In many situations, it will be possible to provide only general information to subjects about the range of uses to which data about them will be put. It is essential that whatever information they do receive is correct and that they are never misled or deceived about those uses. 119. There are two Schedules to the Act—Schedules 2 and 3—that describe situations in which processing is lawful. These relate to personal data and “sensitive” personal data, respectively. However, for sensitive data, both Schedules 2 and 3 must be satisfied. 120. Schedule 2 is satisfied when (among other provisions): (a) either the data subject has given their consent to the processing; (b) or the processing is necessary:

Data Protection Act 1998 | 33



(i) for the exercise of any functions of the Crown, a Minister of the Crown or a government department; or (ii) for the exercise of any functions of a public nature exercised in the public interest; (c) the processing is necessary for the purposes of legitimate interests pursued by the Data Controller or by the third party or parties to whom the data are disclosed, except where the processing is unwarranted in any particular case because of prejudice to the rights and freedoms or legitimate interests of the data subject. 121. Schedule 3 is satisfied when: (a) either the data subject has given their explicit consent; (b) or the processing is necessary (among other provisions): (i) for the exercise of any functions of the Crown, a Minister of the Crown or a government department; or (ii) for medical purposes (including the purposes of preventative medicine, medical diagnosis, medical research, the provision of care and treatment and the management of healthcare services). 122. Under the second principle, disclosure to a third party is allowed as part of further processing only if such disclosure is compatible with the purpose for which the data were obtained. Exemption from the non-disclosure provisions of the Act is also possible in circumstances where the Act recognises that the public interest requires disclosure of personal data, which would otherwise be in breach of the Act. Specific exemptions relate to health, education and social work, and also to research. 123. In Section 30 of the Act, there is provision for personal health data to be exempt from many of the provisions of the Act, subject to specific orders from the Secretary of State. Thus, the way in which the Act regulates health data in registries used for patient care may change with these orders. However, existing rights conferred by the Access to Health Records Act 1990 are likely to be preserved. 124. In Section 33 of the Act, there is provision for exemptions in respect of processing and further processing of data for research purposes provided that the processing is exclusively for those purposes and that: (a) the data are not processed to support measures or decisions relating to particular individuals; and (b) the data are not processed in such a way that substantial damage or distress is, or is likely to be, caused to any data subject. Where this exemption applies, further processing of personal data (i.e. including disclosure) will not be considered incompatible with the purposes for which data were obtained. Data may be kept indefinitely and subject access rights do not apply. These exemptions are not lost if the data are disclosed to any person only for research purposes.

34 | Data Protection Act 1998



The eight data protection principles 1. Personal data shall be processed fairly and lawfully, and, in particular, shall not be processed unless: (a) at least one of the conditions in Schedule 2 is met, and (b) in the case of sensitive personal data at least one of the conditions in Schedule 3 is also met. 2. Personal data shall be obtained only for one or more specified and lawful purposes, and shall not be further processed in any manner incompatible with that purpose or those purposes. 3. Personal data shall be adequate, relevant and not excessive in relation to the purpose or purposes for which they are processed. 4. Personal data shall be accurate and, where necessary, kept up to date. 5. Personal data processed for any purpose or purposes shall not be kept for longer than is necessary for that purpose or those purposes. 6. Personal data shall be processed in accordance with the rights of data subjects under this Act. 7. Appropriate technical and organisational measures shall be taken against unauthorised or unlawful processing of personal data and against accidental loss or destruction of, or damage to, personal data. 8. Personal data shall not be transferred to a country or territory outside he European Economic Area unless that country or territory ensures an adequate level of protection for the rights and freedoms of data subjects in relation to the processing of personal data. 125. To comply with the Data Protection Act 1998 disease registers should if possible obtain explicit consent from data subjects (see section on consent above). It is important that any subsequent processing (including disclosure) is consistent with that consent, with the information provided to patients and with the original purpose for which the data were collected. 126. A registry can comply with the Act without obtaining explicit consent by satisfying one of the other conditions in Schedules 2 and 3. It would seem to us that disease registers could operate within the Act without explicit consent provided that the following conditions are met. (a) Patients in the denominator population should be aware in general terms that health data held about them may be collated by central registers for patient care, healthcare management and research without their explicit consent. (b) Cogent reasons should exist for not obtaining explicit consent (either practical or methodological) and for requiring identifiable data. (c) The work of the register can be shown to be necessary for a legitimate medical purpose that could not be accomplished by some other means. (d) The data controller, and anybody else to whom the data are disclosed, should be a medical practitioner or other professional who owes an equivalent duty of confidentiality to the patient.

Data Protection Act 1998 | 35



127. In addition research registers would comply with the Data Protection Act 1998, and be exempt from some of the provisions of the Act if they satisfy the following conditions. (a) Conditions (a) & (b) in paragraph 125 above. (b) The data are used only for research. (c) The patient’s care is not influenced in any way by the processing of those data. (d) The patient is not contacted by anyone other than their normal professional carer. (e) The patient is not identified in any publications or reports. (f) Data security is of the highest standards. (g) The research makes use of identifiable data only when necessary (e.g. for follow-up, linkage or validation) otherwise anonymous data is used. (h) An appropriate Research Ethics Committee has approved the research, and agreed that obtaining explicit consent is not feasible. (i) The research conforms to any other applicable standards of good research governance—for example, scientific peer review of protocols. 128. Useful advice on the Data Protection Act 1998, and a link to the Act itself, is given by the Information Commissioner’s office at www.dataprotection.gov.uk/ index.htm and by the Department of Health at www.doh.gov.uk/dpa98/ index.htm. Human Rights Act 1998 129. In the Human Rights Act 1998, it states that: (a) everyone has the right to respect for his/her private and family life, home and correspondence; and (b) there shall be no interference by a public authority with the exercise of this right except such as is in accordance with the law and is necessary in a democratic society in the interests of national security, public safety, or the economic wellbeing of the country, for the prevention of disorder or crime, for the protection of health or morals, or for the protection of the rights and freedom of others. 130. Thus, it would appear that any action that involves a breach of privacy must be necessary for the protection of health or it risks legal challenge. Common law considerations 131. There is an important longstanding requirement under common law that, if an individual provides information in confidence, it will be disclosed to others only with the individual’s consent. Confidential information can be disclosed if there is a statutory requirement to do so or if disclosure is justified in the public interest. Unfortunately, only a Court can determine the public interest. Thus, a register that holds data under the conditions of Schedules 2 & 3 of the Data Protection Act 1998 but without obtaining explicit consent may still be at risk of legal challenge under common law. This anomaly in the legal framework is recognised and has led to new provisions under the Health and Social care Act 2001 (see below).

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General Medical Council guidance 132. The General Medical Council (GMC) has issued detailed guidance to doctors about their responsibilities in this area. The full text can be found at www.gmcuk.org/standards/ standards_frameset.htm. The GMC underlines the doctor’s obligation under common law to seek consent before disclosure unless such disclosure is in the public interest. In the guidance, it is emphasised that use of the public interest as a justification can expose a doctor to the risk of legal challenge and GMC action. 133. There are several paragraphs in this guidance on the transfer of information to registries. Although initially it states that doctors “must co-operate by providing relevant information wherever possible”, it is made clear in later sections that: (a) the “express” consent of the patient should be sought wherever practicable before identifiable data can be passed on; (b) patients should be informed about possible future disclosures at “the initial consultation”; (c) if patients object to any disclosures for any reason, only anonymised data should be passed on; (d) whenever patients have been informed about possible disclosures in general, and did not object, but were not asked for express consent to the particular disclosure in question, doctors should themselves assess the likely benefit to the public and “if there is little or no evident public benefit, should not disclose information without the express consent of the patient”; (e) if it is not practicable to seek express consent, doctors must consider whether disclosure would be justified in the public interest, by weighing the benefits of the disclosure to the public health against the possible detriment to the patient. 134. The emphasis throughout the GMC guidance is on individual doctors weighing the public health benefits of each disclosure. This is a cause for concern for registries. Such an approach would undoubtedly lead to variation in practice as different doctors are bound to reach different conclusions. Data—reporting rates would vary unpredictably, and moreover be susceptible to changes in the data protection environment. This could result in the presence of damaging artefacts in the statistics derived from registry data. The stance adopted by the GMC also begs the question whether many doctors have the skills, information, time or interest to make appropriate judgements about benefit to the public health of each and every disclosure. In such situations it would be much easier, quicker and safer for doctors to judge there was no benefit rather than some. 135. The guidance that causes us most concern is contained in paragraph 27, and is as follows: “The automatic transfer of personal information to a registry, whether by electronic or other means, before informing the patient that information will be passed on, is unacceptable save in the most exceptional circumstances. These would be where a court has already decided that there is such an overwhelming public interest in the disclosure of information to a registry that patients' rights to confidentiality are overridden; or where you are willing and able to justify the disclosure, potentially before a court or to the GMC, on the same grounds.”

General Medical Council guidance | 37



136. In the following paragraph, the GMC also states: “Anonymised data will usually be sufficient for clinical audit and for education. …You should not disclose nonanonymised data for clinical audit or education without the patient’s consent.” We disagree. Outcome studies, whether for audit or research, generally require person-based data and personal identifiers. An example would be any study in which the outcomes for surgical units using 30-day mortality rates were compared with those using in-hospital mortality rates. Such a study would require linkage with death data or other outcome data derived from primary care or from death certificates. NHS Policy on confidentiality 137. In 1996, the Chief Medical Officer set up the Caldicott Committee61 to review the transfer of patient-identifiable information for purposes other than direct care or medical research, for example contract monitoring. The purpose was to ensure that information is only transferred for justifiable processes and that only the minimum necessary information is transferred. At the time there was concern that unrestricted developments in IT would lead to widespread dissemination of identifiable data, which could in turn undermine confidentiality. 138. On the whole the committee’s findings were reassuring. They identified 86 separate flows of patient-identifiable information. Although the committee found “no significant evidence of unjustified use of patient-identifiable information” members were concerned that “there was a general lack of awareness … of existing guidance on confidentiality and security, increasing the risk of error or misuse.” The committee also concluded that many of the identifiers currently included could be omitted from transferred information if a reliable coded identifier could be used to support identification. 139. The Caldicott report made a number of recommendations that led to a substantial programme of work to improve policy and practice in this area. Recommendations included the following: (a) Adoption of a set of principles for the prospective approval of all data flows (see below). (b) A programme of work to reinforce confidentiality and data security throughout the NHS (c) Nomination of senior individuals in each NHS organisation to act as “Guardians” in respect of personal data. (d) Much wider use of the NHS number in place of names to render patient data “identifiable” but not "identified”. (e) Restricted access to the NHS number tracing service and other means of identifying individuals from knowledge of their NHS number. 140. In summary, the Caldicott report identified a lack of rigour in the way identifiable data were managed in the NHS and made some straightforward recommendations on how to improve this situation. However, it did not suggest that current uses of identifiable data were unjustified or illegal. The impact of the Caldicott report and subsequent actions, has been a series of measures intended to reduce the risk of a breach of privacy. The committee did not make much comment on the need to restrict access to identifiable data for legitimate purposes. 141. More recently, Ministers have made it clear that Government policy is to obtain

38 | NHS Policy on confidentiality



patients’ informed consent to all uses of identifiable data or to use anonymous data instead. Strong statements have been made, for example in the context of the Royal Liverpool Children’s Hospital enquiry, condemning paternalistic attitudes in the NHS. The Government accepts that this policy cannot be implemented immediately because appropriate anonymous datasets may not be available. As a result regulations are being put in place to ensure that identifiable data can still be used without consent where there is no alternative. However, there is an assumption in Government that, in time, it will be possible to base all surveillance and research work either on data provided with informed consent or on anonymous data. The regulations would then no longer be required. The Caldicott general principles of good practice Principle 1—Justify the purpose(s) Every proposed use or transfer of person-identifiable information within or from an organisation should be clearly defined and scrutinised, with continuing uses regularly reviewed, by an appropriate guardian. Principle 2—Don't use person-identifiable information unless it is absolutely necessary Person-identifiable information items should not be included unless it is essential for the specified purpose(s) of that flow. The need for patients to be identified should be considered at each stage of satisfying the purpose(s). Principle 3—Use the minimum necessary person-identifiable information Where use of person-identifiable information is considered to be essential, the inclusion of each individual item of information should be considered and justified so that the minimum amount of identifiable information is transferred or accessible as is necessary for a given function to be carried out. Principle 4—Access to person-identifiable information should be on a strict need-to-know basis Only those individuals who need access to person-identifiable information should have access to it, and they should only have access to the information items that they need to see. This may mean introducing access controls or splitting information flows where one information flow is used for several purposes. Principle 5—Everyone with access to person-identifiable information should be aware of their responsibilities Action should be taken to ensure that those handling person-identifiable information—both clinical and non-clinical staff—are made fully aware of their responsibilities and obligations to respect confidentiality. Principle 6—Understand and comply with the law Every use of person-identifiable information must be lawful. Someone in each organisation handling confidential information should be responsible for ensuring that the organisation complies with legal requirements.

NHS Policy on confidentiality | 39



Health and Social Care Act 2001 142. Important legislation has been incorporated into the Health and Social Care Act 2001. The new legislation allows the Secretary of State to make regulations covering the processing of patient information for medical purposes, which might otherwise be subject to legal challenge under common law. The regulations can authorise the disclosure of information to any person subject to compliance with conditions. Crucially, the Act specifies that where patient information is processed in accordance with such a regulation, any processing (including any disclosures) required shall be deemed lawful despite any duty of confidence owed by the person doing it. However, the regulations cannot make legal anything that would be illegal under the Data Protection Act 1998. 143. The Secretary of State is required to consult an Advisory Group to be known as the Patient Information Advisory Group (PIAG). All regulations must be laid before Parliament and would only become statutory instruments if approved. The first regulations are to be submitted to Parliament late in 2001. Information on how to apply can be found at (www.doh.gov.uk/ipu/confiden/act/ index.htm). 144. The NHS and the health research community might like to see the new regulations defined in very broad terms. However, if the new powers appear too permissive there is a danger that Parliament will not approve them. The regulations must allow necessary and worthwhile activities to continue but should not remove all meaningful legal safeguards on the use of personal data by health services and medical researchers. In practice there is little danger that the regulations will be overly permissive: the Secretary of State cannot make any regulation that is more permissive than the Data Protection Act 1998. 145. The need for regulations and the way they are operating must be reviewed annually. As information systems develop, and as procedures for obtaining consent and for producing anonymised datasets improve, it may be possible to make the regulations more restrictive by introducing further conditions. However, this may take some time and it seems very unlikely that the need for regulations will ever completely disappear. 146. These regulations are very important for disease registers. In future there will be only two classes of disease register using identifiable data. (a) Those where patients may need to be contacted directly for clinical purposes—these will almost always require patient consent. (b) Those where it is impracticable to obtain consent—these will only be legal if covered by regulations drafted under Section 60 of the Health and Social Care Act. Why do registers need identifiable data? 147. Registers by their nature tend to need personal identifiers to perform their function although some registers need more detail than others. However, there is an important distinction between those registers where patients may need to be contacted directly, for example to invite them for clinical review, and those compiled by third parties that can generally operate using linked anonymised data.

40 | Health and Social Care Act 2001



148. Registers set up with the intention of guiding clinical care normally comprise part of the clinical record. It is inevitable, and desirable, that personal data will be part of the dataset. For example, a general practice-based diabetes register that held anonymous data would be of little value in the recall of patients for regular eye checks. 149. For research registers, where it should never be necessary to contact the patient directly, it may not be immediately apparent why identifiers are required. Indeed, it is often wrongly assumed that most if not all research can be done using anonymous data. 150. Personal identifiers of some sort are necessary for three fundamental "housekeeping” tasks. (a) Avoiding double-counting—To fulfil their function, registers need to be complete. The only way to be sure of finding all cases is routinely to use several independent sources, for example, hospital records and general practice records. As there will be overlap between these sources, duplicates must be eliminated to avoid counting the same people twice or more. Duplicates can be recognised only if patients can be identified. (b) Follow-up over time—To examine causes (past) or effects (future), registers must be maintained over a period of time. Information on past exposures and future outcomes need to be related accurately to registered individuals. This requires some way of knowing whether the person who, for example, died from pneumonia this month was the same person as was registered last year with leukaemia. (c) Validation—If authorities are to act on results derived from analysis of registers, they must have assurance that the register is correct. The most effective way of validating completeness is to compare from time to time the information on the register with another independent source of cases. To do this, cases in both sources must be identifiable in order to know whether the sources agree. Although both sources might have 50 cases, are they the same 50 cases? 151. Identifiable data are also needed when the research question requires linkage between registers: for example, a twin register might be linked to a cancer register, or a general practice diagnostic register might be linked to child health vaccination records. Many important public health questions are unexpected. A single register will rarely hold enough information to answer such a question. One register may contain information on the possible cause (e.g. vaccination records) and another will contain the information on effects (e.g. diagnosis of autism). The only way to find out if the cause is associated with the effect is to look for people who appear on both registers. 152. In practice, the most personal information is used only for initial file building and subsequent additions of data. The named data should never need to be used on a file that also contains clinical data—instead an index number is created within the register and used as a link. However, a certain amount of sensitive data is required for meaningful analysis. For example, age and sex are obviously important, but postcode of residence can also be vital as an indicator of exposure to certain environmental risk factors and as a proxy measure of deprivation. 153. There is currently no system of unique identifiers in the UK that is sufficiently universal to take the place of personal identifiers, such as name, sex, date of birth,

Why do registers need identifiable data? | 41



postcode and mother’s maiden name. In countries such as Sweden, unique identifiers do exist and are used instead of names. In any case, linkage to historical records will probably require names as personal identifiers because it is unlikely that the NHS number will be added to them retrospectively. Why can’t registers always obtain consent before using personal data? 154. Consent from individuals to hold data on a register should be obtained whenever possible. However, there are two general justifications for not obtaining consent: "disproportionate effort”, and methodological problems. Disproportionate effort 155. For the Oxford Record Linkage Study, basic information is obtained from hospitals on about 250,000 hospital admissions per year. It would be impossibly burdensome to contact all those people individually to ask if they had any objection to the analysis of data relating to them. The study has a continuous record of trends in hospital admissions since 1962 for the same population. It is one of the few resources with which It is possible to identify whether statistics that show more hospital admissions mean more people being treated (probably representing better health care) or the same people being treated more often (probably representing poor health care). Methodological problems 156. There is ample evidence that rates of consent may be much less than 100%. Data subjects who do not consent to the use of personal information tend to be different to those who do consent. To obtain sufficiently high response rates, such that these differences can be ignored as a potential source of bias, often involves huge effort. It is likely that few people would refuse to consent to their data being used for public health purposes if the issues were properly explained and understood by them. The problem, however, is that many people are insufficiently motivated to spend time reading information and considering the issue carefully before consenting. Increasingly, people are unhappy to consent to things they feel they do not understand. Thus, non-response is likely to be a much greater problem numerically than negative response. Why are those who do not consent always different from those who do? 157. First, some people are harder to reach by conventional means than others. People who move house more often, or who do not have a home, may not receive letters asking for consent. People who are unwell may not be living at their usual address. People who are blind, who have a learning disability or for whom English is not their first language may not understand a standard letter. Contacting people by telephone may seem intrusive, and will exclude the underprivileged (no telephone) and the deaf in larger numbers than other groups. 158. Second, people vary in their attitudes to unsolicited correspondence or other requests for altruistic actions.62 This depends on the value they place on contributing to the register. People suffering from various forms of disadvantage tend to be more suspicious of "authority” and may not participate for that rea-

42 | Why can’t registers always obtain consent before using personal data?



son. People experiencing some other trouble in their lives (e.g. divorce or bereavement) may be less amenable to participating for that reason. Men and women also have different attitudes to such issues.

"The only correct method of handling persons lost to follow-up is not to have any.” Dorn, 195063

159. As Dorn stated very succinctly, complete participation is important if the research is to produce valid results. If express consent is required to obtain data for registers, they are unlikely to be complete. The most disadvantaged groups in society will be systematically under-represented (e.g. the homeless, disabled, travellers, unemployed, socially deprived, disabled, sick). This may have serious effects including the masking of social inequalities in health. Rather than protecting the interests of these groups, the result would be to make it more difficult to detect and quantify any health problems that affected them.

Why can’t registers always obtain consent before using personal data? | 43


Some Key Questions in Relation to Disease Registers

SOME KEY QUESTIONS IN RELATION TO DISEASE REGISTERS Can the same registers be used to support patient care and research? 160. It is tempting to justify a register by claiming multiple potential or actual applications. However, this is only helpful if those applications are fully served by the register. In practice, each application will tend to place specific demands on the register design and management. For example, the requirements of registers intended for research and those for service uses are substantially different. As a result, there are few registers that fulfil both a meaningful service role and contribute to high-quality scientific studies. 161. For service use, the data should be accurate but completeness is only a partial need. The value of the register is largely proportional to the number of patients covered. Compatibility with other systems may or may not be a requirement. Standardisation of definitions does not much matter provided the register is internally consistent. Patient consent is mandatory but the consent rate is immaterial to the value of the register to those who do consent. 162. For research use, accuracy and completeness are equally important to ensure both the internal and external validity of a register. The number of individuals on the register must be adequate to provide sufficient power in relation to defined questions. Standardisation of definitions is essential to allow interpretation and extrapolation from the research results. Patient consent is desirable but it may not be feasible to seek consent in all cases. If is not practicable to obtain consent, and for other reasons, the register may need to be restricted in purpose to research only to comply with data protection legislation. Table 2: A comparison of some features of service and research registers Data quality Value determined by?

Compatibility and definitions

Consent

Legislation

Funding Costs

Service use Accuracy and timeliness are important Number of cases included

Research use Accuracy and completeness are important Representative group of cases and power in relation to specific research questions Internal consistency is Internal and external consistency important is important to allow extrapolation from results, combined analyses with other registers and interpopulation comparisons Required but non-consent Required but non-consent is a big is only a problem for those problem for the whole register individuals Subjects need access to Access not required but the Data their own information on Protection Act 1998 may require demand that the register is used only for research purposes Various service sources Continuity of funding is important The costs of analysis and reporting should be considered

44 | Can the same registers be used to support patient care and research?



Are registers an alternative to RCTs? 163. The role of registers* in audit is well established. More controversial is the suggestion that some of the same registers could be more widely used in evaluative research and health technology assessment.21,36,64 Results derived from welldesigned and carefully analysed studies using registers have certain advantages when compared with those from randomised clinical trials. First, registers generally contain a more complete sample of cases than clinical trials. The results have greater external validity and may be more useful in clinical practice as a result.49,65 Second, the numbers of cases available for study are often much greater. Finally, historical data sets from registers can be used for retrospective outcome studies that would otherwise take years to achieve prospectively. 164. These advantages must be weighed against the problems of confounding by indication,† protopathic bias or other forms of “allocation bias”: this type of bias is the major methodological drawback of any non-randomised studies of the outcome of therapy.49 Also, registers based on routine data sources may lack the detailed information on co-morbidity necessary to adjust for significant confounders. 165. Despite concerns about non-random assignment, descriptive information from registers can be valuable in certain situations, for example, where randomised studies could not, have not or would not be done.66 Also, statistically precise information on outcomes for specific subgroups is often available only from registers because of the number of observations required. This information can be essential for compiling useful decision analysis models and similar tools for involving patients in decisions about treatment options.21 166. In summary, studies based on registers can supplement data available from randomised studies and should be seen as an important adjunct to rather than a replacement for them. How much do registers cost? 167. To design, set up and maintain a register will incur significant costs, which will vary greatly depending on the nature and scope of the register. Dickinson67 outlined the various components that contribute to the overall cost associated with registers, as follows. (a) Set-up costs, including hardware, software, IT support and training. (b) On-going costs, which may include the full-time salary of an administrator and the costs of routine procedures associated with data collection and preparation. (c) Development costs, for example, upgrading hardware and enhancements to the data collection systems. (d) Private costs, i.e. those incurred by health professionals, patients and carers in contributing to the register mostly in terms of time but also travel, etc. * The term registers is used to avoid confusion but most of the statements in this section also apply to clinical databases. † Confounding occurs when the choice of treatment provided is systematically related to some patient factor that affects outcome, confounding by indication is a special problem in drug studies where treatment choices are influenced by the known or suspected risks and benefits of individual drugs (e.g. women with hypertension are not prescribed the contraceptive pill).

Are registers an alternative to RCTs? | 45



168. Size in terms of numbers of cases registered (or screened) partly determines cost, as does quality. Achieving a high level of completeness requires much effort and, therefore, time. A good quality register generally requires a dedicated administrator and therefore has a minimum cost of around £30,000 per annum. 169. National registers tend to be the most expensive, for example, the national CJD register costs £1.2 million per annum, and a national cardiac intervention database costs up to £500,000 per annum. However some national registers are remarkably economical. The National Osteogenesis Imperfecta Register has been operating since 1980 and is currently managed by one research fellow who spends half a day per week on it. 170. Most modest Regional or District registers cost between £50k and £80k per annum to perform the central functions or the register. Other costs will be incurred in the sites reporting data but these are not normally included in the budget for the register. An economy of scale would seem to apply to registers in that regional registers often do not cost much more than district ones. This may be because a single full-time administrator is needed to run either, and the computing requirements are similar. A typical budget for a regional register is given at Appendix 2. Declared costs for a range of registers are given below. 171. Costs are lower when cases are easy to identify. Patients suffering from some rare conditions will present eventually to a small group of clinical specialists. Data collection from such a small group amongst whom there are existing professional links can be accomplished relatively easily. Active ascertainment is expensive, as is validation of case definitions or validation of the register as a whole. Thus, the set-up and on-going costs of registers that have specially designed systems for collection and validation are expensive. 172. The extent to which the register can be exploited may depend on how much funding is available for analysis and reporting. Substantial costs may accrue if the data are analysed and reported in more than a basic form. Research grants or infrastructure funds are often used to cover the costs of analysis, reporting and dissemination rather than the main register budget. 173. The cost of specialist research registers is highly variable. An idea of these costs can be obtained from the National Research Register. 174. Funding for registers has been insecure in many cases. For this reason, those responsible for running registers have tended to "lose” their costs as much as possible by incorporating their activities into other processes. It is also common for many staff costs to be invisible because they are included in other budgets. Thus, the declared costs of smaller registers are often the marginal costs of the specific activities associated with the register. The true cost of a register may therefore be much higher than that which is declared.

46 | How much do registers cost?



Table 3: Examples of declared costs for Disease Registers (from Survey of DsPH) Description of register Psychiatric case Stroke Cerebral palsy Specialist cancer Arthritis Arthritis register Scleroderma Hepatitis C Coronary heart disease Paediatric diabetes Diabetes Stroke Congenital anomalies

Scope District 3 Hospitals Region National District Region National National District Regional District District Region

Approximate costs (£1000 per annum) 50 91 31 90 150 75 20 60 50 25 for data collection only 15 plus staff costs 80 50

How should registers be funded? 175. Disease registers need stable funding to fulfil their potential. Lead times are relatively long, and substantial costs often accrue before useful outputs can be demonstrated. Lack of a clear mechanism for the funding of disease registers was a common complaint in our survey. Many registers were surviving on small grants from a variety of sources. Much time and energy were diverted into efforts to raise money rather than run the register and report the results. These concerns are not new or confined to this country, as outlined below. "Current mechanisms of support [in the US] are generally poorly suited to providing stable long term funding. Research-oriented databases usually are not supported as databases but rather as part of a plan to answer focused research questions for limited amounts of time. Administrative and research priorities often change more quickly than the database can change to meet those priorities. Consequently, even databases that have successfully met data collection goals can dissolve just as they arrive at the brink of providing answers to the questions they were designed to address.” Pryor et al, 198532 176. To delineate a more appropriate approach to funding disease registers, it is necessary to define the principles underlying any strategy. We suggest the following.

How should registers be funded | 47



(a) Only registers of adequate quality should be funded (i.e. only registers that have been assessed using an acceptable process for evaluation such as the one proposed earlier in this report). (b) Only registers that can address an important health problem should be funded. (c) Duplication of registers should be avoided unless necessary to generate sufficient data. (d) Economies of scale should be exploited where possible. (e) Some non-specific registers should be funded because specific health problems (hypotheses) cannot be anticipated. (f) Rational resource allocation mechanisms should be established at national and sub-national levels because resources are unlikely to be adequate to fund all worthwhile registers. (g) Responsibility for funding individual registers should be determined primarily by the function of those registers. (h) Registers intended to fulfil more than one function should apportion their costs to different funding bodies accordingly. (i) Funding should be awarded on at least a 3-yearly basis, but preferably 5yearly. 177. Bearing these principles in mind, a recommended approach to funding disease registers in England is set out below. Service registers National Service Frameworks 178. Registers to support the national service framework (NSFs) should be funded as part of the clinical service provided by the relevant part of the NHS. For example, practice CHD registers should be provided by practices as part of general medical services. Trusts should supply data on CHD patients to the practices without charge. The same is likely to be true of diabetes registers although the NSF may recommend higher level registers for diabetes. Other service registers 179. Primary Care Trust (PCT) level service registers are likely to be most useful for the planning and evaluation of services and the assessment of needs. They should be funded and managed by PCTs. 180. Regional-level service registers should be funded by Regional Specialist Commissioning Groups (RSCGs) or their successor bodies. The new regional public health departments supporting Government Offices could oversee this process. An example of a regional service register would be a congenital anomaly register. The main function of these is to evaluate screening programmes and provide reliable information on prognosis to inform parents’ decisions. In some cases, national priorities might require regional registers across all regions. If that was the case, to maintain consistency, hypothecated central (or levied) funding should be identified for the purpose. Some national Clinical Audit Databases

48 | How should registers be funded?



may also fall into this category. Those running registers funded regionally should be encouraged to co-operate (within regions) wherever possible, for example, to share staff, infrastructure and data-collection mechanisms. 181. Cancer registries should continue to be funded separately (they have been funded by Regional Offices from April 2001). There has been concern about the lack of a central policy for cancer registers,68 but the situation seems to have improved considerably over the last few years.4 In future, it may even be considered appropriate to expand the role of cancer registries to include other chronic diseases. Bringing together the registration of all chronic diseases at a regional level would make good use of the experience and infrastructure already present in cancer registries. 182. Where it is necessary to set up national service registers, rather than rely on a network of regional registers, these should be funded directly by the Department of Health. For instance, the Human Fertilisation and Embryology Authority Database, the Central Cardiac Audit Database or the proposed national hip replacement register would require a national funding mechanism. In some cases, a national sponsor other than the Department of Health would be more appropriate (e.g. the Medical Devices Agency, Public Health Laboratory Service, or NICE). Research registers “It isn’t easy to obtain support as no one wants to pay for data collection alone but they want the questions answered”. quote from a respondent in our survey 183. Since the publication of Professor Culyer’s report, there has been a drive to identify the costs of research in the NHS and distinguish them from those of teaching and patient care. This principle should be applied to registers wherever possible. 184. Owing to data protection requirements, many registers will increasingly be designated for research purposes only. These registers will then be exempt from some provisions of the Data Protection Act 1998. Registers that are for research only should be explicitly funded for that purpose. 185. Research registers should not be developed in isolation but be part of integrated research programmes in which it is possible to make best use of the registers. Funding for the register should in part be the responsibility of whoever is funding the substantive research programmes. 186. In practice, many funding bodies are unable to justify the strategic objective of funding a register when it is set against discrete research projects. For this reason, a mechanism must be found to support registers as a form of research infrastructure that does not put them in competition with individual project proposals. Capital grants are currently allocated separately by funding bodies for buildings, laboratories and major equipment in response to bids and after peer review. Bids are judged on their scientific quality, relevance to strategic aims and the likely productivity of the group over the long term. There would seem to be no reason why grants for registers could not be allocated in a similar way.

How should registers be funded? | 49



Nationally important research 187. The major research sponsors (including the Department of Health) should be invited to contribute to a central “non-capital” health research infrastructure fund administered by the Department. Bids for disease registers and related proposals of the highest scientific quality, the contribution of which was likely to be of national or international significance (not necessarily national scope), could be made to this fund. Registers funded by this mechanism should comply with the principles of good practice set out in this report and elsewhere. Successful bidders would be awarded long-term support on a rolling basis with regular reviews. Any NHS support costs incurred by these registers (for example, clinical staff abstracting data) would be covered by the Support for Science allocations to health-care providers. Specific applications 188. Appropriate sponsoring bodies should fund those registers that have specific research functions. For example, the Medical Devices Agency should continue to fund registers that are intended to gather information about the safety of medical devices. Health Technology Assessment might be another example. There are also several specialist research charities that could be invited to fund relevant registers. Local research 189. Local collaborative research groupings may choose to use their Priorities and Needs funding to support local disease registers or to develop research programmes associated with local service registers. Local research registers might be regarded as pilots, which could be used as a basis for bids to the central fund in due course.

“In our experience the funding of registers is considered a peripheral exercise with little or no support given for the development of research based on the data accumulating. The data on this register is a world resource and could be developed in many ways. However the NHS considers it purely as an audit exercise and will not support the research aspects.” quote from a respondent in our survey

What is the national role in respect of registers? 190. Although a devolved approach to disease registers is helpful when responding to local interests, experience and needs, the current situation suffers from a lack of central co-ordination and a lack of consistency in policy. There is also a need to set and monitor standards for registers nationally. Finally, registers need to have a role in the new national strategy for public health research.

50 | What is the national role in respect of registers?



“[For clinically-rich databases] engendering enthusiasm, and co-ordinating the processes, will (we believe) long remain a task requiring special skills and expertise, and central funding.” Lilford, Braunholtz and Richardson, 199964 191. Central co-ordination of policy and practice is important. A national “register of registers” would be useful in assessing the need to develop new registers or to continue to fund old ones. This could be maintained by the public health observatories. This mechanism/system would also make the registers more accessible to potential users. 192. Establishing a consistent national approach to funding registers is perhaps the next most important task. The approach described above would allow the value of individual registers to be assessed at an appropriate level and should ensure a fair outcome in terms of resource allocation. 193. Another urgent priority is to clarify the data protection requirements for registers. The Secretary of State should propose regulations under Section 60 of the Health and Social Care Act that would allow the use of personal data for important disease registers without explicit consent where necessary. 194. Where networks of regional or district registers are developed, there will be a need for central collation and analysis of the data. This function, and that of developing and monitoring national standards for disease registers, suggest the need for some form of academically based national co-ordinating centre for disease registers. National Co-ordinating Centres 195. To promote a common set of standards for disease registers, national funding should be sought for one or more disease register co-ordinating centres (along the lines of the successful clinical trials centres). These centres would have several functions: (a) to develop methodology in relation to disease registers; (b) to provide advice to research groups; (c) to develop data standards for common disease registers; (d) to provide practical support to service registries (e.g. designing questionnaires, providing data definitions); (e) to undertake quality assurance for local registers; (f) to collate information from a network of selected local service registers to provide a national dataset. Information for Health 196. Another function for the national centres could be to advise on how disease registers might relate to work in support of the Information for Health strategy. This would be

What is the national role in respect of registers? | 51



a mutual interaction. First, developing NHS information systems need to be designed in such a way that disease registers can continue to operate and use the data from electronic health records. Second, disease registers can provide a mechanism to achieve many of the objectives of the strategy if they are used appropriately. Research registers 197. Arrangements for NHS research are currently being transformed in line with the document Research and Development for a First Class Service. Further changes are in train as a result of the abolition of Regional Offices and Health Authorities. Research registers are likely to be supported under both the Support for Science and the Priorities and Needs R and D programmes. Research sponsors such as the MRC and Wellcome Trust should be invited to collaborate with the Department of Health and the NHS in developing a strategic approach to disease registers. For example, work is already underway on the UK Population Biomedical Collection.* The aim of collaboration would be to ensure common standards, avoid duplication of effort and encourage sharing of (anonymous) data for research. 198. The Research Analysis and Information Directorate of the Department of Health is currently reorganising its central funding schemes. The funding of registers should be included under the programme for funding of research infrastructure and capacity building. The Department’s Policy Research Programme is developing specific proposals along these lines. The role of the MRC 199. The MRC have funded a number of registers in the past and are committed to supporting the development of high-quality registers for the purposes of research. The MRC provided the following statement for the purpose of this report. “MRC believes that disease registries offer valuable research opportunities, particularly when linked with other health, economic, social and demographic data (for example data held by Public Health Observatories). Whether disease registries are to be overseen by a central co-ordinating body or are to be managed within a distributed model, it is important that there is some strategic overview of the creation, maintenance, quality assurance and utilisation of disease registries—a process in which MRC would be happy to participate. There will be metho-dological/technical challenges in quality-assuring and using registry data, linking it to other pertinent data, and analysing such large datasets. MRC is thinking about the challenges of data linkage in relation to the proposed UK Population Biomedical Collection and the disease-based Cohort DNA Collections. It would be helpful to the Council to be able to develop its plans in co-ordination with the Public Health Observatories and other partners. The Council would also be interested in funding innovative public health research projects drawing on registry and other data.” * The UK Population Biomedical Collection will be a repository of DNA samples, lifestyle data, and medical records for approximately 500,000 consented UK adults.

52 | What is the national role in respect of registers?

Results of the National Survey of Directors of Public Health Disease Registers in England

RESULTS OF THE NATIONAL SURVEY OF DIRECTORS OF PUBLIC HEALTH 200. We wrote to all district and regional directors of public health (DsPH) in England asking for details of local population-based registers, emphasising our interest in the conditions set out in Saving Lives: Our Healthier Nation. We also asked for their general views on the value of disease registers in public health practice. To encourage responses, we enclosed copies of recent Clinical Standards Advisory Group reports, which we thought would be of interest to the recipients. We received replies from 61 of 99 Districts, a remarkably high response rate (62%) for this kind of survey. Moreover, most replies were thoughtful and specifically addressed the questions we had posed. We also contacted the Regional Directors of R&D in England. Views of Directors of Public Health 201. One of the most complete responses from a district DPH is summarised below. In this district, there was an active district-wide diabetes register but there were no plans to develop centralised district registers in other areas until the Electronic Health Record was fully developed and could be exploited. , Diabetes: District-wide register since 1996; 8,000+ individuals registered; only 1or 2 patients have declined to be included; all practices and all clinics participate; run on an Access database that links to hospital information systems. The register is used for call/recall for eyes and podiatry. There is a full-time co-ordinator for the register based in a Trust. Mental health: Several general practices have registers of severe and enduring mental illness. The local A&E department keeps records of all suicide attempts and self-harm in electronic format. One Primary Care Group has established a sophisticated electronic disease register for mental illness. Cardiovascular disease: No plans for a district register. All practices are working towards NSF CHD registers (standard 3). Templates are being developed across the district to ensure compatibility. With the introduction of the EPR we hope [eventually] to have a virtual CHD register across the NHS. The local hospital maintains a stroke register. Regional registers: Cerebral palsy and cystic fibrosis run by the local academic centre. Response from a District respondent 202. A reasonably clear pattern along these lines emerged from the replies of district DsPH. albeit with some variation. By far the most commonly reported districtwide register was a diabetes register (n=23), many of which were run by Trusts on behalf of the health authority. Only a handful (n=5) of district-wide CHD registers were reported.

Views of Directors of Public Health | 53

Disease Registers in England Results of the National Survey of Directors of Public Health

“We have an active, and now almost complete, diabetes register in the district, which is run by our local acute Trust but is community-based in terms of coverage. It has been based on the Salford Register model and we [the HA] participate actively in user group involvement in the development of that database. Our current strategic issue locally is whether to migrate from a stand-alone register, such as the Salford system, to an electronic patient record as described in Information for Health.” Response from a district DPH “There are to my knowledge no accident or mental health registers in [our District]. All local PCGs are committed to developing CHD registers as part of their contribution to the HImP. In practice, however, progress is patchy and I could not honestly claim that local registers are up and running. There is a good diabetes register covering the whole district, capturing data from general practice, hospitals, optometrists and chiropodists and linked to the deaths register for regular updating. I believe disease registers are essential—not just for public health but also for the practical management of chronic disease.” Response from a district DPH

203. The respondent from one public health observatory (PHO) provided us with complete data on diabetes registers in their region. Results from our (incomplete) national sample were consistent with the information from the PHO: of 11 districts, 2 had district-wide diabetes registers, 2 had hospital-based registers and 1 had a comprehensive set of primary care-based diabetes registers. 204. Respondents from many districts reported a policy decision not to develop centralised registers but instead to support the development of practice-based registers. In other districts, structures were being set up to allow better access to GP data for ad-hoc purposes.

“We have a current project to support data quality in local practices within a clinical governance framework. We believe it is best to place registers as close as possible to where the patient care takes place. In this way there is a greater chance that the registers will be kept up to date and used for call and recall, clinical management and audit. I believe registers are essential if we are to improve the quality of care for people with major chronic disease. However, our experience in [District] is that such registers do not need to be centrally based.

54 | Views of Directors of Public Health


As with most other Districts, there is a good deal of work going on around improving our ability to access GP-based information in a consistent way in order to support the new National Service Frameworks on heart disease and mental health. However, I am not aware of established registers in the areas of Saving Lives.” Response from a district DPH

205. Several district DsPH were concerned about the quality of registers based in general practice, as exemplified by the quotation below. “While 70% of practices in our district are reported to have coronary heart disease registers, I suspect there is little science to these and we are, therefore, having to rework them, together with the practices, to try and develop a consistent district-wide approach.” Response from a district DPH

206. One response shows that local public health departments may not know what registries are collecting data in their own district. This emphasises the need for co-ordination of effort and for communication on disease registers.

“I have talked to my colleagues, and there has been an acute outbreak of head scratching and thinking in relation to the questions in your letter. This is because it is hard to be sure about all developments across the district and region, as well as within NHS Trusts and primary health care teams. There are also differences of professional view on the value of disease registers.” Response from a district respondent

207. One DPH pointed out that our letter implied a medical model of registers, which forms only part of the public health interest in relation to registers. “A number of my team work in multi-agency public health where the sorts of population registers they work with are of those in defined client groups, or receiving care at particular levels, etc. A number of registers of this type come to mind: Child health / Care Programme Approach (Serious Mental Illness) / Child protection / Disability / STD / Children involved in prostitution”. Response from a district DPH



208. A substantial group of district DsPH were supportive of registers in principle but were concerned that they should be of good quality and well run to be of any use. There was an implication in most of these responses that many of the current registers do not meet implicit required standards. “Disease registers are only of value to public health if; (1) they are validated and regularly updated; (2) clinically relevant (e.g. for recall, screening or clinical management); (3) appropriately resourced and supported; (4) accessible to appropriate staff; (5) secure and confidential; (6) consistent with the evidence base.” Response from a district DPH

209. Support for registers both for service use and as a research tool was widespread. However, there was genuine scepticism over the benefit to public health of a comprehensive system of disease registers for common diseases, even in districts that were fairly advanced in their use of registers. Some districts had registers but were not using them for public health purposes. “Using this philosophy [clinically-relevant registers for CHD] we have found direct benefit in areas such as: • setting and monitoring clinical governance targets; • benchmarking practices and monitoring overall quality; • targeting strategy for secondary prevention (risk stratification). [However,] I have never been of the belief that these kind of data are particularly strong for so-called “needs assessment“. Whilst we probably generate as good community-based prevalence data as anybody in areas such as hypertension and ischaemic heart disease, such information has no practical use in predicting service needs or demands. Highly specialised service or patient registers might fulfil this role, but for most common conditions ”text book“ prevalence figures are just as useful.” Response from a district DPH “[Our] doubts focus on two questions. Is there a solid research basis to show that the establishment of ”a series of disease registers around the country“ really will deliver better health? If disease registers are costly, what is the opportunity cost of this work in relation to other researchbased options for improving the population’s health?” Response from a district respondent

56 | Views of Directors of Public Health


“Disease registers are expensive to set up and difficult to maintain. My own personal view is that they are useful in terms of rarer conditions, but I have never been convinced in terms of more prevalent conditions.” Response from a district DPH “I have never been very keen on registers, chiefly because of the considerable effort to establish and maintain them. At local level I have always thought their main value was to provide a mailing list to keep patients informed about local policy or advances in treatment. On the research front I was impressed by [research project] which would have been impossible without his accurate and updated disease register.” Response from a district DPH

210. Other respondents were more enthusiastic, but remained concerned that registers should be established with a clear purpose and be designed to support clinical practice or epidemiology in a tangible and specific way. These respondents pointed out the need for population-based registers for public health purposes. 211. Other respondents were generally in favour of registers in principle but felt that they had little to offer public health in practice. When considering the uncertain benefits and high costs of registers, several respondents thought they would represent poor (or at best uncertain) value for money. “Registers are one way of improving our understanding of morbidity and I would suggest that proposals to create registers need to be seen in that context. To some extent a register is a ”gold standard“ that may be unrealistic where improvements in routinely collected morbidity data may be more achievable (and affordable) in the short to medium term.” Response from a regional public health respondent “I have to say that local disease registers have not been very valuable to the Public Health function in [our district]. I think there are a number of potential benefits particularly in relation to coronary heart disease, diabetes and cancer but there are also significant opportunity costs; it is only in relation to the cancer register that we have achieved usable data.” “A number of Primary Care Groups have put effort into establishing practice based coronary heart disease registers with very limited success. This is partly due to IT limitations but is mainly due to the difficulty in changing culture and practice to ensure consistent, complete and valid data recording on disease areas.” Response from a district DPH



212. A significant minority of DsPH seemed to be ignoring/disinterested in the public health role of registers. They appeared happy to leave the issue of disease registers to clinicians in Trusts and general practice. 213. Concern about data protection and confidentiality issues was mentioned by some respondents as a practical obstacle to the future public health use of registers. 214. There was strong support for basing disease registers on the Electronic Patient / Health Record. “I feel strongly that disease registers should be a product of electronic patient records and not ”stand-alone“ entities. I am concerned that if too much effort goes into producing registers the main vision of EPRs will be diminished.” Response from a district DPH

215. There was a preponderance of more unusual disease registers in teaching hospital districts. These were often run by academic units based in a clinical setting and which had both research and service functions. Many of them were not population-based but relied on the hospital’s catchment population to provide cases. 216. Despite the range of opinions expressed above, the majority of responses acknowledged the importance, whether potential or actual, of disease registers for public health. “I believe that health improvement can be gained as much from aggregating up good personal care in a systematic way as delivering any other grand scale public health schemes. In order to achieve this I think disease registers have an important role to play. I will certainly be putting a lot of emphasis on this within our own district for the future.” Response from a district DPH

Views of Regional Directors of R&D 217. Most regional R&D directorates have funded disease registers for research purposes in the past and some still do. Those who do not support registers believe the R&D Levy should be used to fund only those projects using data held by registries, not the registers themselves. In general, there was strong support for disease registers but dismay at the lack of structure in relation to commissioning and quality assurance.

58 | Views of Regional Directors of R&D


“My problem with disease registers has been a lack of clarity about their purpose. Some seemed to have been curiosity driven by interested parties, some to provide the basis for service provision, some to study the natural history and some for more formal research purposes. Many have lacked quality assurance. In all the funding arrangements have been insecure.” Response from a regional Director of R&D “Disease registers constitute an important research resource. However, we feel that more could be done in a strategic context to improve the co-ordination and selection of appropriate registers. We also feel that more could be done to facilitate legitimate access to the data held by registers.” Response from a regional R&D respondent

Views of Regional Directors of R&D | 59


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National Leprosy Registry of Norway from 1856 until today. Int J Epidemiol 1973 Spring; 2 (1): 81–9. Doll R, Muir C, Waterhouse J. Cancer incidence in five continents, 2. Berlin: Springer-Verlag, 1970. Hill RD. Community care service for diabetics in the Poole area. Br Med J 1976 May; 1 (6018): 1137–9. Charlton BG, Taylor PRA, Proctor SJ. The PACE (population-adjusted clinical epidemiology) strategy: a new approach to multi-centred clinical research. Quarterly Journal of Medicine 1997; 90: 147–51. Alberman E. Disease registers. In: Armitage P, Editor. Encyclopaedia of Biostatistics. Vol. II. Chichester UK: John Wiley, 1998: 1180–5. Hall S, Bellman M. Reye's syndrome in the British Isles: first annual report of the joint British Paediatric Association and Communicable Disease Surveillance Centre surveillance scheme. Br Med J (Clin Res Ed) 1984 Feb; 288 (6416): 548–50. Hall SM. Reye's syndrome and aspirin: a review. J R Soc Med 1986 Oct; 79 (10): 596–8. Newton L, Hall SM. Reye's syndrome in the British Isles: report for 1990/91 and the first decade of surveillance. Commun Dis Rep CDR Rev 1993 Jan; 3 (1): R11–6. Mathiesen EB, Ahlbom A, Bermann G, Lindgren JU. Total hip replacement and cancer. A cohort study. J Bone Joint Surg Br 1995 May; 77 (3): 345–50. Measles, MMR, and autism: the confusion continues. Lancet 2000 Apr; 355 (9213): 1379. Skegg DC. Hormone therapy and heart disease after the menopause. Lancet 2001 Oct; 358 (9289): 1196–7. Britton A, McKee M, Black N, McPherson K, Sanderson C, Bain C. Choosing between randomised and non-randomised studies: a systematic review. Health Technol Assess 1998; 2 (13): i–iv, 1–124. Goldacre MJ, Kurina LM, Seagroatt V, Yeates D. Abortion and breast cancer: a case-control record linkage study. J Epidemiol Community Health 2001 May; 55 (5): 336–7. Thornes R. Case registers in 1999. London: Royal College of Paediatrics and Child Health, 2000. Vaughan N. Personal experience with a district diabetes register system—DIALOG—linked to the FHSA database for co-ordinating diabetes care: design and implementation. In: Dawson A, Ferrero M. Chronic Disease Management Registers: proceedings of a workshop. London: HMSO, 1996: 99–120. Gill L. Methods for automatic record matching and linkage and their use in national statistics: national statistics methodological series No. 25. London: National Statistics, 2001. Medical Research Council. Personal information in medical research. London: MRC, 2000. Yawn BP, Yawn RA, Geier GR, Xia Z, Jacobsen SJ. The impact of requiring patient authorization for use of data in medical records research. J Fam Pract 1998 Nov; 47 (5): 361–5. Klaucke D, Buehler J, Thacker S, Parrish R, Trowbridge F, Berkelman R. Guidelines for evaluating surveillance sytems. MMMR 1988; 37 (S5): 1–18.

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Appendix 1—Design of the Database

APPENDIX 1 DESIGN OF THE DISEASE REGISTERS DATABASE Field

Notes

ID

Simple identifier

NAME

Name of the register

STATUS

Clinical Database or Disease Register, if Disease Register this field is also used to show whether data collection is ongoing, patchy, terminated or not yet started

ABBR

Commonly used abbreviation if any

INDEX TERM

One of 28 categories of registers (e.g. stroke, CHD, diabetes, haematology, congenital anomalies etc.)

SCOPE 1

Specific description of the subject of the database (e.g. cerebral palsy)

SCOPE 2

National / regional / district

CASE DEFINITION

Full description of the precise case definition used

APPROX CASES

Latest estimate of number of cases registered

LOCATION

Where the data are held

CONTACT SURNAME

Self explanatory

CONTACT ID

Identifies the contact in a relational database containing addresses, numbers, e-mails etc.

INTERNET ACCESS

Internet address relating to the register

POPULATION COVERED

As much detail as possible on the size and nature of the population contributing cases to the register

START DATE

Date data collection started

RESEARCH

Whether the register has been used for research and details of any publications produced

FUNDING

Specific sources of funding

COST

Estimated annual cost of running the register

NOTES

Any additional information including methods of ascertaining cases and the extent to which the register has been validated

SOURCE

How we became aware of the register

64 | Appendix 1

Appendix 1—Design of the Database


Fields for recording the results of an evaluation of the quality of the register* SENSITIVITY TIMELINESS REPRESENTATIVENESS PREDICTIVE VALUE ACCURACY / COMPLETENESS SIMPLICITY FLEXIBILITY ACCEPTABILITY * See Klaucke et al (1988) for a detailed description of how to assess each attribute Example of a database entry ID NAME ABBR STATUS INDEX TERM SCOPE1 SCOPE 2 CASE DEFINITION APPROX CASES LOCATION CONTACT SURNAME CONTACT FORENAME INTERNET ACCESS POPULATION COVERED POPULATION SIZE START DATE FUNDING COST RESEARCH SENSITIVITY TIMELINESS REPRESENTATIVENESS PREDICTIVE VALUE ACCURACY/COMPLETENESS SIMPLICITY FLEXIBILITY ACCEPTABILITY NOTES

SOURCE STAGE

76 HCV National Register Disease register / ongoing Hepatitis C Hepatitis C National A transfusion recipient traced during the HCV look back exercise who tested either positive or indeterminate for antibodies to HCV 871 (out of possible 997 traced) PHLS Communicable Disease Surveillance Centre, Colindale,London Harris Dr Helen Transfusion recipients identified by lookback exercise 997 01-Jan-98 DoH £60k pa. 3 yr projected costs £82k pa yes

Register currently being extended to cover other infections with known acquisition date. Annual follow up of cases. Cases flagged within the NHS Central Register. Annual call for study proposals. NRR 3

Appendix 1 | 65


Appendix 2—Budget for a Typical Register

APPENDIX 2 BUDGET FOR A TYPICAL UNIVERSITY BASED REGIONAL REGISTER Salaries 1 WTE Administrator (Grade II.8) 0.5 WTE Secretary (Grade C.4) Total salaries

Year 1

Year 2

Year 3

Total

33646 8743 42389

36159 9049 45208

37425 9366 46791

107230 27158 134388

2000 300 500 1300 600 700

1500 300 500 1300 600 700 650 1000

1600 300 500 1300 600 700 675 1000

5100 900 1500 3900 1800 2100 1325 2000

5075

6000 19525

Consumables Travel & Subsistence Telephone Stationery Printing notification forms Postage Computer consumables Computer maintenance Report production Database software, installation, training Total consumables

6000 9400

Equipment Computers (2) Printer Answerphone Fax machine Office furniture Total equipment

4300 500 150 520 1600 7070

5050

4300 500 150 520 1600 7070

Summary Year 1 Salaries 35010 Employer’s NI and superannuation 7379 Equipment 7070 Expenses and other costs 9400

Year 2 37313 7895

Year 3 38618 8173

5050

5075

Total 110941 23447 7070 19525

Sub total costs

58859

50258

51866

160983

Overheads 40%*

16956

18083

18716

53755

TOTAL COSTS

75815

68341

70582

214738

66 | Appendix 2

Appendix 3—Diabetes Registers


APPENDIX 3 DIABETES REGISTERS 1.

Diabetes registers are probably the archetype of chronic disease registers. Most diabetics remain so for life and have continuing health, and health-care needs. Information from registers and elsewhere suggests that the impact of diabetes at the population level may have been underestimated. Diabetes care requires effective audit and diabetes registers have been shown to be feasible at practice and at district level.

2.

Support for the establishment and maintenance of diabetes registers comes from both international policymakers (World Health Organization and the International Diabetes Foundation) and national groups (British Diabetic Association and the Clinical Standards Advisory Group).1 Although direct empirical research evidence of their effectiveness is lacking, clinical consensus in favour of diabetes registers is strong. From 1990 onwards, firm recommendations have been made describing diabetes registers as essential for the provision of effective diabetes care and to monitoring the outcomes of that care (see below). Diabetes care should be structured, and identifying all diabetic patients is a fundamental step in ensuring that patients receive the care they require.

Some recommendations on diabetes registers from UK authorities CSAG, 19941 Patient, locality and service-based registers are “essential in assessing quality of care”. Registers should show: — the service (doctor) responsible for the diabetes care of that patient — year of diagnosis — the date of last annual review for complications screening. Locality-based registers are most useful in assessing whether diabetes care is reaching all those with the condition. Service-based registers are essential in assessing quality of care. Locality-based registers are essential for identifying rates of default from care and deaths. Clinical Outcomes Group, 19972 The St Vincent Declaration targets and definitions should be at the heart of local target setting, outcome measurement and monitoring. A district population-based register is a way of undertaking this monitoring. British Diabetic Association, 1991 A comprehensive diabetic register, at practice, clinic and district level, and their aggregation, is an essential prerequisite for collection of outcome data to monitor progress towards St Vincent targets. Information on ethnicity is needed in routinely collected data.

Appendix 3 | 67



Role of registers in diabetes care (based on Home, 1996) 3.

Several of the characteristics of diabetes itself are relevant to the role of diabetes registers. (a) It is progressive and is associated with serious "late" complications, therefore management needs to be active and continuous. (b) Some forms of diabetes are complicated to manage requiring many different skills and specialist services applied in a co-ordinated fashion. (c) Most continuing care of diabetes should be preventive requiring clinicians to initiate contact rather than respond to symptoms (by which time it may be too late). (d) Preventive care and surveillance for early signs of complications are cheap when compared with the cost of treating established complications. (e) Care is provided in both primary and secondary care settings, but the relative distribution of workload varies considerably from place to place. (f) There is robust evidence that the care provided often falls short of what could be achieved in terms of prevention, screening, communication and education, particularly in areas where information systems are weak.1 Diabetes registers can be used to improve patient care in the following ways. Quality assurance—review of achievements by comparison with agreed targets for process measures (eye screening), outcome measures (limb amputations), risk factor reductions (smoking and blood pressure), and intermediate clinical outcomes (blood glucose control or lipids). Recall for surveillance—this is useful in two forms; (1) eye screening for retinopathy; (2) annual recall for more general review. Needs assessment—people with diabetes consume about 5% of all health-care resources, and the prevalence of diabetes can vary considerably among populations.

4.

A useful diabetes register should have the following characteristics. (a) The dataset must be compatible with the UK modified DiabCare dataset3 to allow aggregation with other registers. This probably also requires the use of a structured care record in the clinical notes for ease and accuracy of abstraction. The standard precise definitions for each datafield should be used. Incomplete submissions of partial datasets can still be useful provided standard definitions are used. (b) Quality assurance software should be incorporated to provide feedback to contributing clinical units on achievements against targets (see above). (c) There must be a recall facility linked to an up-to-date demographic register, which could be either the practice registration file or the main district administrative register.

68 | Appendix 3



(d) The register should be flexible such that it is possible to incorporate any developments in diabetes datasets. 5.

Specialist hospital diabetic units were in general the first to develop “registers” of their diabetic patients.4,5 However, in the last decade, general practice-based diabetes registers have been widely adopted. Hospital-based registers tend to be more detailed in the information collected and are often used for research as well as patient care. Like other hospital-based “registers”, they are rarely population based.

6.

Much of the emphasis in the care of diabetes is on the prevention of complications and on the identification of any such complications as early as possible. There is evidence that many of these functions can be carried out in primary care. For this reason, a practice-based approach to registration seems attractive. In 1993, only 20 of 89 practices approached by a group of London GPs had a diabetes register;6 by 1996, over 95% of all practices in England had registers.7 This extraordinary growth was partly a result of developments in practice information systems, but was also driven by the Department of Health’s policy on chronic disease management.

The Chronic Disease Management Programme 7.

The Chronic Disease Management Programme originated in 1990, but was further expanded in July 1993. It provided for additional payments to practices at which registers of diabetic patients and/or patients with asthma were maintained.7 Registers also had to be used by practices to carry out annual reviews, and audit, and to produce annual reports for the Health Authority (then the Family Health Services Authority—FHSA). There were no stipulations in the programme about how the information should be stored or validated, or how information from other sources should be incorporated into the registers.

8.

The deficiencies of the registers set up in response to the Chronic Disease Management Programme could have been predicted. The registers were highly variable in their format and quality. Aggregation across practices was attempted but was difficult due to incompatibility of systems and data specifications. Moreover, validation between practices and hospitals was not part of the scheme, leading to double-counting and incomplete identification as well as duplication of effort across primary and secondary care.8

District-wide registers 9.

To realise the potential benefits of diabetes registers, it was recognised that data from overlapping primary and secondary care registers should be collated to produce complete population-based registers. This would provide Health Authorities in particular with useful information on the local epidemiology of diabetes and patterns of service use.9 Such registers could also be used to assess the quality and equity of care provided in terms of both process and outcome.

10.

For administrative convenience, the obvious level at which to attempt this aggregation was the Health District. The first district-wide register was set up 25 years ago10 in Poole. It has been successfully exploited to monitor trends in diabetes prevalence over time.11 However, in 1996, only about 10% of Districts claimed to have “district population-based registers”7 and many of these were incomplete. The results of our survey suggest that the current figure is nearer

Appendix 3 | 69



20% although quality and completeness vary enormously. Aggregation has also been attempted for some hospital-based registers and by some GP Medical Audit Advisory Groups. Special software programmes such as DIALOG have been developed which can provide the IT functions required.12 A report of the implementation of DIALOG stressed the importance of a full-time register manager in successfully introducing the system.13 11.

The construction of an effective district-wide register is not a trivial exercise.6,14,15 The following activities were required to set up and maintain the diabetes register in Lanarkshire.16 (a) Initial data entry (some 10,000 prevalent cases identified from diabetic clinics and GP practices)—in one hospital some data were downloaded from a hospital database, in two hospitals one consultant with the aid of an audit assistant loaded all the hospital cases, in another hospital a team of wardbased nurses helped to abstract retrospective data from clinical notes. Funding for clerical support was provided for the general practices. (b) For ongoing data collection part-time staff are employed in the hospital clinics and practice nurses conducting diabetic clinics enter most primary care data although an audit facilitator is required to visit some practices to abstract data from clinical notes. (c) It took four years to achieve a prevalence of 2.08% on this register suggesting almost complete ascertainment.

12.

The Lanarkshire register disclosed low rates for all process measures for patients managed by GPs when compared with those for patients visiting hospital diabetic clinics. For example, visual acuity had been measured in 85% of hospital patients but in only 28% of GP patients. Clearly this register will be a very useful tool in stimulating and monitoring improvements in these measures.

13.

However, Elwyn and colleagues have questioned the value of district-based registers over and above that of practice-based registers.17 They identified the following problems for district registers. (a) Consent and confidentiality—there is increasing acceptance that disease registers used to drive patient care require patient consent for the transfer of personal data to a third party and should comply in other ways with the “fair processing” principle. This is a considerable additional administrative burden on the register and risks incomplete and biased ascertainment. (b) Practical difficulties—to be helpful, district registers need to be accurate, complete and continually updated. This requires the regular transfer and integration of data from primary and secondary care, and possibly those from other sources. Although theoretically attractive this is difficult to achieve due to the prevailing state of NHS information systems. Despite the existence of a national standard dataset, there is wide variation in the data actually collected. (c) The cost-effectiveness of district registers is largely unknown.18 It probably costs at least £60,000 per year to maintain a district register. There are opportunity costs to take into account, and there are also, hidden costs for example, time contributed by members of the data committee. The effectiveness of registers appears to be improving both the quantity and quality

70 | Appendix 3



of monitoring in primary care and thereby improving intermediate clinical outcomes such as glycosylated haemoglobin.19 The overall question remains whether achieving hospital levels of control in primary care is a worthwhile objective given the differences in the risk profiles of the populations concerned. Validity and completeness 14.

In theory, for District registers, it is possible to obtain data from several sources such as optometry services and blind registers. However, many of these sources contribute very few new cases and in practice the greatest yield is obtained from general practice and hospital datasets. General practice data can include prescription information as well as diagnostic data. There is considerable debate about the benefits of integrating data from secondary care and other central sources via record-linkage as opposed to relying on primary care data and the normal communications between hospitals and GP practices.20,19

15.

The prevalence of diabetes is known from the National Health Survey for England (1991–1994) to be at least 2% (see Table) yet early attempts to compile diabetes registers were regularly reporting prevalence figures between 1.0% to 1.2%.15 Although much work is necessary to reach levels of completeness that give prevalence rates approaching 2% in general practice registers, it would now seem to be achievable.19 One factor may be that the underlying prevalence of diabetes is increasing.19,21

16.

The results of a study in which record-linkage was used to construct a diabetes register from central sources (hospital discharges, attendances at an eye screening clinic, pathology results) showed that this method identified 96% of diabetics and had a positive predictive value of 95%. This register was slightly more sensitive than the GP registers.20

17.

In Oxford, a generic linked database was constructed using data from eight local general practices and the hospital discharge data already held in the Oxford Record Linkage Study (ORLS). The practice prescribing data, routine diagnostic codes in GP records and hospital diagnostic data were used to identify treated diabetics. From these data, it was easy to calculate the prevalence of diabetes in this population. The Table below shows the results in comparison with the equivalent information from the National Health Survey for England (1991–1994).

Table: Prevalence of diabetes: results from a linked GP and hospital database compared with those from the Health Survey for England (1991–1994) Age group (years)

20–39 40–59 60+ Total

Males

Females

England

ORLS—GP

England

ORLS—GP

0.5% 2.5% 6.2% 2.6%

0.5% 2.2% 5.6% 2.2%

0.4% 1.6% 4.7% 1.9%

0.7% 1.7% 4.2% 2.0%

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18.


Despite the attractions of district-based registers, the problems faced during their establishment and use cannot be dismissed. Although the technical difficulties may be overcome soon, the data protection issues are becoming more important than ever. The experience of constructing coronary heart disease registers in support of the National Service Framework is likely to be instructive. A similar approach might be effective for diabetes registers.

Datasets 19.

A common dataset, and an agreed approach to its use, are crucial if register data are to be amalgamated. The WHO DiabCare dataset was developed to monitor the St Vincent Declaration targets. However, this dataset was considered to be too demanding to collect in Britain, and the British Diabetic Association and the Royal College of Physicians have recommended a modified version.3,7 The UK modified DiabCare dataset has three levels of entry but does not conform to the Read coding system used in many GP practices. Only the first level seems to have found favour with British GPs.

Integration with clinical information systems 20.

Most diabetes registers have been set up as bespoke disease-specific systems, often as part of more generic computer systems in general practice. When the Department of Health’s strategy, Information for Health, has been fully implemented, the Electronic Patient Record may provide the ideal vehicle for populating a diabetes register as a more automatic by-product of normal clinical work. Further guidance on this is likely to be provided in the National Service Framework for Diabetes.

Consent and confidentiality 21.

Any register that relies on the transfer of personal data beyond the normal professional carer, and which might influence the care that the patient receives (for example, by precipitating recall for screening), should seek the express consent of the patients concerned. However, the maintenance of a practice-based register, identifiable data from which are not submitted to any other agency, would seem to us to constitute part of normal clinical record-keeping and should not require any special consent. Nevertheless, it is good practice, and an important part of the principle of the “fair processing” of personal data, to ensure that all patients are fully informed about all uses of their personal data. They should be made aware of how and why data about them are used in as much detail as is appropriate in the specific context.

22.

It is also good practice to form a data ownership committee or other steering or reference group to review all uses of information derived from a register. Health authorities do not need access to identifiable data for the purposes of needs assessment and service review. Where record-linkage is required to eliminate double-counting and to add follow-up information, it is important that identifiers are used only for the linkage processes, and are stripped from the main files used for analysis. Research registers may need more freedom to use identifiable data for external linkages and for subsequent follow up studies. Any such work should have ethics committee approval (see also section on confidentiality in the main report).

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Diabetes registers and research 23.

The aim of most diabetes registers is to improve the care of registered patients. Some of these registers may also be used for research, particularly as a source of names and addresses of patients who can be asked to take part in specific studies. Others are specifically designed with research in mind.

24.

An example of a diabetes register that was set up primarily for research purposes is the Yorkshire Children’s Diabetes Register.22,23 Information is collected on all children (