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Diagnostic and screening programs for fetal anomalies and genetic disease: how should we evaluate them? Baird, Patricia A. Apr 30, 1994

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Centre for Health Servicesand Policy ResearchDIAGNOSTIC AND SCREENING PROGRAMSFOR FETAL ANOMALIES AND GENETIC DISEASE:HOW SHOULD WE EVALUATE THEM?Patricia BairdHPRU 94:4D April 1994Health Policy Research UnitDiscussion Paper SeriesTHE UNIVERSITY OF BRITISH COLUMBIACENTRE FOR HEALTH SERVICES AND POLICY RESEARCHDISCUSSION PAPER94 - 4DDIAGNOSTIC AND SCREENING PROGRAMS FOR FETALANOMALIES AND GENETIC DISEASE:HOW SHOULD WE EVALUATE THEM?Patricia Baird, M.D.,C.M.,FRCPC.,FCCMG.The Centre for Health Services and Policy Research vas .stablished by theBoard of Governors of the University of British Columbia in December 1990.It vaa officially opened in July 1991. The Centre's primary objective 1sto co-ordinate, facilitate, and undertake multidisciplinary research inthe areas of health policy, health services research, population health,and health human resources . It brings together researchers in a varietyof disciplines who are committed to a multidisciplinary approach toresearch, and to promoting wide dissemination and discussion of researchresults, in these areas. The Centre aims to contribute to the improvementof population health by being responsive to the reaearch needs of thoseresponsible for health policy. To this end, 1t provides a researchresource for graduate students; develops and facilitates access to healthand health care databases; sponsors seminars, workshops, conferences andpolicy consultations; and distributes Discussion Papers, Research Reportsand publication reprints resulting from the research programs of Centrefaculty.The Centre's Health Policy Research Unit Discussion Paper aeries providesa vehicle for the circulation of preliminary (pre-publication) work ofCentre faculty and associates. It 1s intended to promote discussion andto elicit comments and suggestions that might be incorporated within thework prior to publication. While the Centre prints and distributes thesepapers for this purpose, the views in the papers are those of theauthor(s).A complete list of available Health Policy Research Unit Discussion Papersand Reprints, along with an address to which requests for copies ahould beaent, appears at the back of each paper.2DIAGNOSTIC AND SCREENING PROGRAMS FOR FETAL ANOMALIES ANDGENETIC DISEASE: HOW SHOULD WE EVALUATE THEM?I was asked to talk about evaluating diagnostic and screening programs for fetal anomalies;I'm going to take this to include genetic diseases as well, as they are so closely related. I amgoing to talk about programs that are aimed at detecting prenatally whether the fetus has a.genetic disease or a physical anomaly. I am not going to address newborn or other screening.I will tell you what we have learned from Commission research about how prenatal programsare currently used in Canada, talk about developments on the horizon, and mention some ofthe issues raised by such programs. I will conclude my talk by describing the usual approachto evaluating prenatal diagnostic and screening programs, pointing out some of thedetrimental effects of this approach, and outlining an alternative.What programs exist in this country?I. Prenatal Screenine PrommsThe prenatal screening tests that are in use currently in Canada are shown on the left ofFigure 1. If an abnormal result is revealed in one of them, the woman is simply at a higherrisk of having an affected fetus and she is then offered one of the diagnostic tests on the rightof the figure.a) Screening Tests on Maternal BloodSeveral screening tests can be done on maternal blood; the most frequently used of these isMaternal Serum Alpha Feto Protein (MSAFP) in which a protein produced by the fetus ismeasured. A higher than normal level of it in the pregnant woman's blood suggests thepossibility of an abnormal fetal opening, a neural tube defect. Concentrations of AFP alsorise in some other fetal conditions - so that although a high result means that there is anincreased likelihood of neural tube defect, it may also indicate other problems. More recentlyit has been shown that a low level of MSAFP may indicate a chromosome anomaly.Triple Testing is another maternal blood screening test and relies on a combination of threedifferent indicators in the pregnant woman's blood sample, one of them being AFP; it can beused to detect the likelihood of Down Syndrome.With all these blood tests, a cut off point must be used to offer diagnostic testing that willresult in identification of an optimal number of affected fetuses, without subjecting aninordinate number of women to unnecessary diagnostic testing.It is very probable that population screening by one of these methods will replace thematernal age criterion for eligibility for amniocentesis for chromosome anomalies. Researchlooking for other biochemical indicators is going on, as is research on where to mostappropriately fix the cutoffs.3b. The second screening test is "routine" ultrasound scanning. It is important torecognize that ultrasound scanning can be used as a screening test, but when it is used in amore intensive, and specialized way, looking for something specific, it is also a diagnostictest. Most pregnant women in Canada now have "routine" ultrasound scanning to estimategestational age; to see if there is more than one fetus; and to look for conditions that mayrequire medical attention. In this process, ultrasound scanning may produce images that raise. the suspicion of a congenital anomaly. I want to emphasize that -although this type ofscanning does pick up some of the more major malformations such as anencephaly, it is not areliable way to diagnose fetal anomalies. A normal routine scan does not give muchinformation regarding this aspect of the fetus.II. Prenatal Dialnostic PrommsWhat about prenatal diagnostic programs, which are offered only to women at higher risk inCanada?a) amniocentesis is normally done between 15 and 17 weeks of pregnancy. The samplemay be examined biochemically for the level of substances such as AFP but it also containsfetal cells. These cells may be grown in culture and examined in different ways; forexample, to analyse the chromosomes, or to examine the DNA, so exactly how the sample istested depends on the indication for diagnostic testing.b) The next prenatal diagnostic test is chorionic villus sampling or CVS. CVS can bedone several weeks earlier in pregnancy than amnio, but it may be somewhat more difficultto interpret the results. It also cannot be used to diagnose some physical abnormalities suchas neural tube defects. It is most useful for single gene disorders.c) The third diagnostic test is diagnostic ultrasound which involves a specialized detailedexamination of the fetus, and it can accurately diagnose many congenital anomalies.m. Future DevelopmentsWhat is coming in the future? The discovery of the polymerase chain reaction (PCR), whichcan amplify the DNA from a cell or two to produce amounts large enough to do diagnostictests on, means that there are two potential developments in the future. One is to test fetalcells that occur in maternal blood; and the other is with regard to preimplantation diagnosis.i) Testing of Fetal Cells in Maternal BloodDuring the first trimester there are perhaps two to six fetal cells per million maternal cells inthe woman's circulation. If these could be sorted and identified, then it is theoreticallypossible, because of PCR, to identify any single gene disorder by examining fetal cells fromthe maternal circulation. It is also possible to see if these fetal cells have a chromosomaltrisomy.There are many technological problems to sorting fetal cells from maternal blood and to4testing such small samples, but they are being worked on currently. In future these kinds ofdevelopments may prove feasible, be able to be automated and thus be feasible on apopulation screening level.ill Preimplantation DiagnosisThe other technique that is on the horizon is preimplantation diagnosis. This technology. involves removal of one or a few cells from an early human 'zygote obtained after IVF,examining the cells for a particular genetic disorder and only transferring the zygote to theuterus if it doesn't carry the disorder.IV. Use of prenatal diamostic tests in CanadaI'd like to outline something of what we learned from Commission research about the use ofprenatal diagnostic and screening tests in this country, starting with diagnostic tests. Thereare' 22 genetics centres in Canada - they provide diagnostic prenatal tests - amnio, CVS andtargetted ultrasound - usually in association with a specialized radiology/ultrasound unit.In 1990 in Canada we found that over 22,000 women were referred for prenatal diagnostictesting because of an identified higher risk. This represents about 6% of all women who gavebirth in Canada in that year.Figure 2 shows the reasons they were referred. A substantial majority, 78 %, were for olderage - over 35 years. The remaining 22 % were referred for a variety of reasons, including aprevious child with a chromosome abnormality, or family history. Some were referred afterabnormal results on screening tests.Not all women who are referred choose to have diagnostic testing, and some women don'thave the test because fetal death is discovered, or they miscarry before testing. But themajority of referred women - over 89% - have diagnostic testing.Of the almost 20,000 women tested in Canada in 1990 about 95% received reassuringresults. A fetal disorder was detected in the remainder of cases - about 990 women. Of these,792 women, or 80%, decided to terminate the pregnancy. So that when a serious fetaldisorder is detected by diagnostic testing, overall about four out of five women in Canadadecide to terminate the pregnancy and about one woman in five decides to carry on with thepregnancy. To put this in perspective less than one in a hundred terminations in Canada aredone after PND. ,As figure 3 shows the decision to terminate after a fetal disorder is detected varies with thenature of the disorder. The disorders in the last two groups listed have less serious or lesspredictable effects than disorders in the other groups. The fact that fewer women choose toterminate a pregnancy when one of these less severe disorders is detected shows that womenand couples consider carefully the severity and burden of the disorder before making adecision. Data from studies on other less severe disorders or those with later onset, for5example, polycystic kidney disease, show very few couples decide to have an abortion whenthe condition is very variable and treatable, and few even want PND. It is evident that thedecisions of women and couples are nuanced and situation specific.It is important to realize that social pressures or lack of support or services for the disabled. are not the only, or even the primary, factor in the decision for many women. Althoughthere is no question that increased support and services for individuals with disabilities arenecessary in the interests of social justice, having them would not provide an acceptablealternative to PND and abortion of affected fetuses for many women and couples. To say thatit would, neglects the devastating impact of some mental and neurological disabilities, whichrequire life-long care, often overwhelm the parents' lives and inflict suffering on the affectedindividual and, as a result, on family members and others who witness that suffering. Inaddition, parents - especially the woman, who often bears the primary responsibility for care- are left with few choices about pursuing other goals.As the tables I've shown you demonstrate, it is possible to give fairly good statistics on theoutcome from these kinds of diagnostic testing in this country. With regard to the outcome ofprenatal screening tests - it is not so easy.IV• Use of Prenatal Screenine Tests in CanadaPrenatal screening tests are done by a variety of practitioners widely dispersed in thecommunity, and a very large number are done. In total there are several thousand generalpractitioners, obstetricians and radiologists across Canada who may provide routineultrasound or take MSAFP samples, and good data are simply not available.The kind of information provided to women by practitioners prior to screening testing variesgreatly. This is true even in the one province, Manitoba, where a provincial MSAFPprogram has existed. In a survey of Manitoba practitioners done for the Commission, only30% of them passed on written information they were provided to the woman before takingblood, 54% provided only oral information, and 6.6% provided no information at all.Another finding was that only 79% offered the test to all pregnant women - over 6% did notoffer it to any.The other common screening test - routine ultrasound scanning - is now a frequentcomponent of prenatal care, though opinions differ on the number of ultrasounds that shouldideally be done during pregnancy, and indeed on whether it is a good use of resources toperform ultrasound scans routinely. Routine ultrasound scanning during pregnancy is anexample of a medical technology that entered wide-spread use without demonstratedeffectiveness. The present state of knowledge leaves some room for differing opinions, butstudies have failed to show a measurable reduction of obstetrical complications or betteroutcomes associated with routine ultrasound scanning in pregnancy. Consensus conferenceshave produced a variety of opinions on routine ultrasound in pregnancy. There is no questionthat routine ultrasound can help date pregnancies and detect multi-pregnancies - but there is6no data to show this makes a difference on a population basis in terms of birth outcomes.Yet at the same time, ultrasound screening costs enormous amounts of money; there is asignificant opportunity cost from using the money in that way. Despite the lack of evidenceregarding its effectiveness, there has been a massive increase in the number and cost ofultrasounds done in Canada over the last 10 years as Figure 4 shows. Commission research. found the bulk - about 75% - of this increase is attributable to an increase in the per capitause of routine ultrasound, rather than an increase in the number of pregnancies. Clearly it isessential to determine whether routine ultrasound is in fact effective in managing pregnanciesmore safely and effectively, since knowing this would have important implications forresource allocation and for medical practice.If routine ultrasound does not help manage pregnancies more safely and effectively, then thecurrent number of tests is obviously too high. If routine ultrasound does achieve thispurpose, however, the distribution of tests is seriously skewed, because some women arenever offered screening: we found the percentage of all pregnant women having anultrasound examination ranges from 97% in Quebec - where they usually have two - to 62%in Manitoba.It is essential to evaluate this rapid proliferation of routine ultrasound and to determinewhether the substantial funds now being devoted to it are justified. This is a majorundertaking, since a trial powerful enough to detect clinically significant effects might requirea sample size of over 12,000. Nevertheless, evidence from major multi-centre randomizedcontrol trials would help to determine the effectiveness and value of routine ultrasound as aprocedure to help manage normal pregnancies.Routine ultrasound scanning is not a reliable way of screening for congenital abnormalities.Only major structural abnormalities (such as anencephaly) are reliably detected. Targeteddiagnostic scanning, on the other hand, done because a woman has been identifed as at riskof an anomaly, if it is carried out by a skilled and experienced operator, is very valuable.The efficacy depends on location and the type of the anomaly but it is quite high - forexample for neural tube defects. However most malformations are unexpected and there is noreason to do diagnostic targeted ultrasound on most women.Screenine and the Need for PrOI!l"8JD Frameworks"Routine" ultrasound in pregnancy badly needs a program framework that would serve tocontain appropriately both utilization rates and total costs. A Commission study of ultrasounduse rates in British Columbia and in Ontario over the last 10 years showed clearly that theB.C. approach, in which ultrasound can be provided only by licensed hospital facilities and ispaid for out of hospital budgets, is far more effective in containing use and costs than is theOntario system, which does not restrict practice location. Ontario physicians With ultrasoundequipment in their offices can bill for prenatal ultrasounds they decide are needed and thatthey conduct in their own offices. In the nine years up to 1991, expenditures for prenatal7ultrasound increased twofold in British Columbia but fourfold in Ontario. While the numberof hospital-based ultrasounds increased by only 16% in Ontario between 1983-84 and 1989­90, the number of non-hospital ultrasounds increased by about 300%. The British Columbiaapproach not only appropriately controls costs but makes possible quality control andstandards of training for personnel. It also eliminates the potential for self referrals, whichhave been shown in many studies of medical practice to lead to unnecessary use.With regard to MSAFP screening - with the exception of a couple of provinces - the use ofthis is growing, marked by an absence of program guidelines and inconsistency in follow-upcounselling. This proliferation of MSAFP testing outside established programs is worrisome.Commission research shows that the indications for which MSAFP screening tests are offeredvary dramatically across the country. In some locations, it is being offered routinely byphysicians; in other locations, depending on the physician it may be offered to all or only tothose at higher risk. In locations where a genetics centre analyses the MSAFP samples, thecentre also does follow up counselling. But in other cases, particularly where privatelaboratories analyse the samples, counselling is left to the general practitioner or obstetrician,who may not have the knowledge or experience to provide appropriate counselling. As withroutine ultrasound, these variations reflect a lack of clear guidelines and standards forproviding MSAFP screening and counselling.Given the problems associated with the proliferation of MSAFP, it seems more appropriatethat it should be used only as a routine screening test where there is a program funded bygovernment, that includes well-designed information for women, education of physiciansabout the program, and the necessary facilities and resources to ensure accurate interpretationand follow-up, including counselling, when the results are abnormal. Quality control andmonitoring of service delivery is essential to protect those using the services: governmentshave a responsibility to ensure that ad hoc provision does not occur.The decision about whether to offer MSAFP, triple testing or other blood screening tests ona population basis is complex, particularly as the technology is changing rapidly. Also recentinformation about increased levels of folic acid in the diet of pregnant women reducing therisk of neural tube defects needs to be taken into account in decisions about whether and howto extend testing more widely.In essence, if population screening is to be offered routinely, I believe it should be in thecontext of properly designed programs within the publicly funded health care system.TechnoloeY Assessment and Future Evolution of PrommsWe can assume that the pace of development of knowledge that could be applied in prenataltesting is not going to lessen. The desire for information about the fetus in higher-riskpregnancies, together with continuing scientific discovery, is likely to produce a steadystream of new developments. It is essential, therefore, to put in place appropriate technologyassessment.8Ideally new technologies should not be made available except in the context of clinical trialsuntil their safety and accuracy have been assessed. Canada has been a leader in the field ofclinical trials for prenatal diagnostic techniques before their introduction to clinical practice.The assessment, introduction and use of both amniocentesis and CVS have constituted goodexamples of technology assessment and evidence-based medicine. . For example, a. collaborative multi-eentre trial in 1976, supported by the MRC, was carried out to assess thesafety and effectiveness of amniocentesis, and it helped establish international clinicalstandards for its delivery; the first multi-centre randomized clinical trial comparing CVS withamniocentesis was completed in Canada, and was made possible by a voluntary agreementamong all Canadian genetics centres that CVS would be available only within the context ofthe trial.In short we found that the main categories of prenatal diagnostic tests are used in the contextof written protocols, by highly trained personnel and with data collection - and thesetechnologies had national multi-centre trials early in their history. The track record in Canadafor assessing new prenatal diagnostic technologies at the centres has been good.In comparison, routine ultrasound and MSAFP screening have simply proliferated, ratherthan being assessed and then introduced. Why have these screening tests been allowed toproliferate without proper assessment? They are non-invasive, they are relatively easy toadminister - no special expertise is required to draw a blood sample or even to performroutine ultrasound, and the procedures pose few immediate risks to the pregnant woman orthe fetus. As a result, many thousands of physicians in Canada could, if they wanted,provide these tests. By contrast, fewer than 100 medical professionals work at the 22genetics centres. Needless to say, it is much more difficult to control the introduction anddissemination of new techniques when practitioners are so dispersed.But all tests should be examined against the same stringent results-based criteria regardless ofthe extent of invasiveness and immediate risk. The use of non-beneficial technologies subjectspeople to unnecessary procedures, furthers inappropriate medicalization, and uses resourcespoorly, with substantial opportunity costs.Further, the fact that a test is non-invasive or easy to administer says nothing about the oftenextensive laboratory and analytic resources required to provide meaningful results oradequate counselling about them. Worrisome for the future is if the analysis of fetal cells inpregnant women's blood proves effective. Unless appropriate policy is put in place, thiscould mean that blood samples are taken widely without the resources and expertise forinterpretation and follow-up counselling.The new PND technologies being developed vary in risk and invasiveness. It is likely thatsome of the more invasive, risky and expensive tests (such as preimplantation diagnosis) willreceive the same thorough assessment as amniocentesis and CVS before being introduced, butit is likely others will not, unless we make some changes in our approach.9A more formal process of technology assessment could help to .ensure that funding isavailable to conduct trials needed to produce the information on which practice can be based.For example, a large multi-centre trial of routine ultrasound would cost several milliondollars. However, given that up to $100 million is spent annually in Canada on these tests, itis important to determine whether it should continue to be offered routinely.To date, the MRC has funded the clinical trials that have been done in the area of prenataltesting. However, the MRC does not have enough money to fund expensive new trials.Provincial ministries are responsible for funding and managing the health care system, andtechnology assessment is part of that responsibility, so the burden of funding trials willincreasingly fall on provincial ministries of health. However, not all provinces are able tofund their own clinical trials, and in any case this would lead to unnecessary duplication.This means a process that would set priorities and co-ordinate the funding of clinical trials inthis area would be of benefit.Some Issues Raised by Prenatal Screenine and DiaenosisThere are many issues raised and although not strictly my topic, I'd like to touch on a few ofthem briefly, because they do have relevance to evaluating programs.One of the issues is that PND may impinge on the autonomy and choice of pregnant women,and that there may be either coercion to have the tests, or with-holding of them. The extentto which this occurs will affect the status of women in society and the equality of the sexes.With regard to autonomy and choice by women, the results of a nationwide survey ofphysicians done for the Commission are relevant. This showed that 15% of physiciansopposed abortion after PND no matter how serious the congenital condition, and that at theother extreme a similar percentage thought it irresponsible for women at identified higherrisk not to have PND. On the question of who should decide which fetal anomalies justifyabortion, 40 % of referring physicians believe it is the .physician, not women or couples.These results show that a disturbing proportion of referring physicians do not endorse theprinciple that patients should make their own informed choice and this has a substantialimpact on access to PND services. Correlating the pattern of referrals with the pattern ofresponses from a national survey of physicians, as well as from the Canadian population, wefound the more serious doctors in an area consider congenital anomalies to be and the morethey approve of the option of termination, the more likely they are to extend access and referto a centre. The variation was over four fold. This is of concern as respect for pregnantwomen's autonomy requires that it be her values and priorities, not the doctor's, thatdetermine her decision to accept or decline testing.Attitudes to People with DisabilitiesAnother issue raised is whether the availability of PND will lead to discrimination against10people with disabilities and contribute to their marginalization. Some people are alsoconcerned that society's perception of what constitutes a serious disorder will change and thatPND will over time, lead to greater intolerance of even minor anomalies. It is important,therefore, to say something about what constitutes a 'serious' disorder. A disorder can beserious because of the suffering it causes for the child and/or because of its emotional, .physical and financial effects on the parents and family. These vary from one disorder toanother.Tay-Sachs, for example, is a particularly severe disorder in terms of the suffering it causesfor the child; affected children have short and painful lives. Down syndrome can beburdensome for parents; though such children may lead long and happy lives, they requireconstant care. If congenital heart disease is also present, Down syndrome can cause severesuffering for the child, but even in cases where the child's symptoms are not so serious, thechild is usually incapable of functioning independently.Other conditions cannot be considered serious because they cause neither significant sufferingto the child nor hardship for the parents, for example, extra digits. Children with such minoranomalies do not suffer ill health from them, and caring for them does not create unusualhardship for the parents. Allowing PND and termination for such disorders would beunethical for several reasons. First, it reflects inappropriate views of disability, of the respectowed to human life, and of the nature and value of children and the parent-child relationship.It also violates the principle of appropriate use of resources, since it fulfils no real medicalneed. It is important that PND continue to be used only to detect serious disorders.It is not possible, however, to establish a definitive list of serious disorders. The severity andimpact of a disorder can change over time, with changes in society and in the treatability ofdisorders.We see no evidence that Canadian women, genetics centres or provincial governments areusing PND for trivial disorders. This is an important issue, however, and one about which atolerant and inclusive society must remain vigilant.Diversion of ResourcesAnother concern expressed by people with disabilities is that providing resources for PNDmay divert money away from programs providing support to families and people withdisabilities. Adequate social support for parents bringing up a child with a disability isimportant, both as a matter of justice for people with disabilities and as a matter of informedchoice for the parents. This support is essential if the decision to continue a pregnancy andhave a child with a disability is to be a viable option. I do not believe it is an either/orsituation of providing PND or providing support for people with disabilities. On the contrary,the two go hand in hand, even in the same family.11Attitudes toward people with disabilities are changing as society becomes more informedabout their needs and more aware of how constitutional and human rights' protections arerelevant. Increased attention to the needs of people with disabilities is being paid by all levelsof government. This is in the same time period that PND has become available. Thus, theconcerns expressed about PND need to be placed in this context of positive changes inCanadian policies and institutions with respect to disability. .Prejudice or hostility toward people with disabilities is unacceptable; the question is whetherthe existence and continuing development of PND technology promotes such attitudes. I donot accept that it does. I believe it is possible to uncouple the issue of the availability of PNDfrom the issue of society's attitude toward and treatment of people with disabilities.All couples face the possibility of having a child with a disorder even if the risk is low formost. But the commitment to value and nurture an existing child with a disability is not thesame as the commitment to nurture a fetus where an anomaly has been detected. Lack ofcommitment to continue a pregnancy does not mean that commitment to an existing child orperson is diminished.Some people wishing to have PND are already caring for an affected child. They usuallycare deeply about their child, and many have struggled to improve social support andacceptance for people with disabilities. However, they may feel unable to cope with raisinganother affected child, or not feel they should have another child suffer in the same way, andso they seek PND for future pregnancies. Many couples at risk wish to avoid having anotherchild with severe disabilities. To assume that this desire represents hostility or prejudicetoward existing children or adults with disabilities is, I believe, an oversimplification, bothmorally and psychologically.To argue that the status of people with disabilites would be improved if PND were lessavailable is misleading. Given that most disabilities are not congenital and cannot bediagnosed prenatally, it seems likely that society's approach to people with disabilities willnot stand or fall on the availability of prenatal diagnosis services. There need not be anyconflict between the interests and needs of couples at risk and those of people withdisabilities. To suggest that Canadians should choose to support only one or the oilier is anexample of an unnecessary adversarial stance.Canadians' Attitudes to PND and AbortionCanadians' attitudes toward abortion are complex, but in the Commission's survey ofCanadians, most support a woman's right to choose in such circumstances. Most Canadiansrecognize that the diagnosis of a severe congenital anomaly in a wanted pregnancy is tragicand that it is with great regret that a woman or couple decides to terminate the pregnancy.Most Canadians do not feel they can tell others what they should do in these circumstances.About three-quarters say that if the fetus has a severe anomaly, the parents should have theoption to terminate the pregnancy . Canadians recognize that termination in this situation is12not a benefit but rather the opposite. Public support for having this option available tocouples at risk is therefore a case of public respect for the extremely difficult situation ofthese couples - a situation in which all the options are difficult and none of the choices iseasy.PermittinK Life with a FamilyIt is also important to consider the situation faced by couples at higher risk when PND is notavailable. The desire to have children is deeply rooted, and most Canadians want to havechildren. Where PND is not available, a couple's knowledge that they are at higher riskposes a serious threat to this goal.Couples who want families are willing to take the usual risks that accompany reproduction.But, for example, when couples know they have a 25 % risk of a genetic disorder, unlessPND with the option of termination is available, most couples give up their plans to havechildren (or more children) rather than risk having an affected child. This is often deeplydistressing to the couple, particularly if it is their first and only child who is affected.There are many other issues raised which I don't have time to go into but which are alsorelevant to evaluation of prenatal diagnostic and screening programs, for example protectionof privacy and confidentiality, informed choice, equity in access, safety and efficacy. Butfrom the data that I have outlined and from survey research done for the Royal Commission,it is evident that most Canadians want to be able to avoid the birth of a seriouslyhandicapped child. The availability of PND for severe disorders and the freedom of womenand couples to choose among options including termination, seem to them a medical serviceworth supporting. Provided PND diagnostic and screening programs for severe disorders aredelivered with unbiased, accurate information and fully informed consent, PND is bothbeneficial to individual women and couples at risk, and consistent with social valuesregarding equality for persons with disabilities. It is evident that a goal important to mostCanadians is to have a healthy family; and that a cost paid by higher risk couples to reachthat goal is to have termination of an affected fetus andtry again.Axmroaches to Evaluation of Prenatal PrommsThis brings me to the way prenatal diagnosis programs have usually been evaluated. In thepresent situation of constrained resources for medical care, genetics program directors andothers have tended to do program evaluations that are really directed to justifying adequatefunding for programs; I have done it myself. The approach is that there are costs and thereare benefits which are added up in dollar units - the financial cost of a PND program iscalculated, often exhaustively and carefully, and then this is expressed as cost per birthavoided. (Figure 5) Because securing funding for PND services was usually the goal behindsuch analyses, it is perhaps understandable that they use dollars as the sole unit ofmeasurement and ignore other costs and benefits as not being able to be measured. Theanalyses compare the cost of prevention by screening and prenatal diagnosis, with the cost of13treating a patient. They show that prevention is cheaper.Framing the approach this way gives a message that what is really important is that caringfor the handicapped among us is costlier than preventing their birth. This framing of theissue is very upsetting to patients and their families, and sends a message to the public thatphysicians, geneticists and health planners are lacking in compassion. It implies that caring. for patients is not what the medical system is for, but to save money. But most medicalservices are not expected to pay for themselves by saving costs. It is not suggested, forexample, that only enough cholecystectomies be done that avoid the costs of not doing them.A solely monetary cost-benefit approach is hurtful to families as well as to the affectedindividuals themselves, since it describes patients and families as a financial burden, ratherthan as equal and valued members of society. It is particularly threatening because peoplethey rely on - physicians and those responsible for health care resource allocation - frame itthis way. To treat prevention and care as either/or also makes the public concerned, andraises fear of misuse of genetic knowledge and technology, given the history of past abuses.It is a bad approach also because it obscures the facts and misrepresents reality. Savingmoney is in fact not why most physicians and health policy planners support preventionprograms. Prevention is not better because it is cheaper; it is better because it allows peoplebetter and more fulfilling lives and avoids suffering. It is better because it allows people achoice in something that is a very important part of their lives - having a family. It allowsthem the choice of avoiding an unforeseen or unwished birth of an affected child. In practice,if a couple decide to terminate an affected pregnancy, they almost always go on to haveadditional healthy children. This is an emotional, social and economic benefit which isneglected in this approach.Non financial costs and benefits have traditionally been excluded because they are extremelydifficult to measure objectively - but it is precisely these intangible benefits that are mostimportant to people, and that are what the provision of medical care is about. If we are tocontinue to be a caring society, it is essential we consider the non-financial costs and benefitsto individuals, to families, and to the larger society in our evaluation of programs and in ourplanning.In essence, simply looking at aspects that can be measured in monetary units has seriousconsequences. We need a more encompassing approach to evaluation of prenatal programsthat more accurately reflects the real issues to patients, physicians and funders of services.The analysis of a medical service must be based on the objectives of relieving suffering andengendering health. A common framework is needed for describing the true costs andbenefits of prenatal screening and diagnostic programs - a framework which should take intoaccount the objectives of medical care. As well it is possible to factor in non-financial costsand benefits in a more objective way, and I'd like to touch on this too.14A Broader Approach to EvaluationDrummond has said that a cost-benefit analysis must include:1. Describe the service and alternative services or policies2. List all the identifiable financial and non-financial costs and benefits of these policiesfor the patient, the family and society.3. Measure these costs and benefits in a way that is useful for comparing differentpolicies.4. Evaluate the costs and benefits of different policies in equivalent units wherepossible. 1This means valid, non-financial units of measurement are needed as well as dollar ones. Acost benefit analysis approach has been used by Bernadette Modell to Thalassemia - which isthe beginning of such a new perspective." I'd like to describe it because it is an approach thatcould be adapted to evaluation of other prenatal programs as well, and it is a way that betterreflects the caring ethos of health care.Figure 6 outlines an assessment of the costs and benefits of Thalassemia screening andprenatal diagnosis in Cyprus. The framework includes both treatment and prevention,recognizing their complementarity. It makes overt that there are costs and benefits at everystep. You can also see that accepted, affected children are counted as a benefit in thisapproach to evaluation, as is replacement to achieve a healthy family.The benefits of such programs are informed people; informed choice of at-risk couples; andthe birth of healthy infants or of accepted, affected infants. These are the major justificationfor service, not that they offer financial savings or conserve resources for other purposes.The proportion of parents who make an informed choice to have an affected child will differ;the severity and treatability of the diagnosed disorder will largely determine this. Since thecost of treating severe chronic disease is so great, such programs may well save money, butthis should not be the primary objective.Equity in health and health care is a goal we all espouse, and it should also apply with regardto congenital anomalies .and genetic diseases. We know that certain sub groups within thepopulation are at higher risk. To give those individuals the same chance as other people tohave a healthy family, prenatal programs are needed. A major benefit it provides is for thoseindividuals to have the same chance as others to have a healthy family.Objective MeasurementsAs well as a common expanded framework which allows a more holistic approach toevaluation, there are objective, non-financial measures that can be used in evaluating the15outcome of such programs. Since at risk couples do not simply want to avoid having affectedchildren but they want to have a healthy family like most other couples, there are objectiveunits in which to measure whether they are disadvantaged or not in doing this compared tothe general population. It can be measured as follows:1. Average family size of at-risk families compared to the general population families.2. Proportion of affected children to unaffected children in the at-risk families comparedto general population families.3. The percentage of wanted pregnancies aborted in the families compared to the generalpopulation.In Cyprus without any programs in place, at-risk couples were at about 75% of thepopulation norm for unaffected to affected children since a quarter of their children were ill.When only counselling was available then such couples dropped much lower than others interms of family size because so many of them stop reproducing. When population screeningand prenatal diagnosis became available, such couples were found to be almost as likely tohave the same number of healthy children as the general population. These parameters can bemeasured objectively.What has haggened to birth grevalence where there are gro&rams for other conditions?In the whole of the U.K. between the late sixties and mid-eighties, the birth prevalence ofanencephaly has decreased by 94%. About half of this fall may have been due to a decreasein background incidence; but half can be definitely attributed to MSAFP screening andselective abortion.' In our country - in Manitoba - since the MSAFP program was introducedin 1987 there has been a 50% decline in incidence of liveborn infants with neural tubedefects.In short, as well as being able to assess objectively the outcomes in affected families, it isclearly possible to monitor outcomes in a population that are attributable to prenatalscreening and diagnosis programs. Following changes in birth incidence in conjunction withthe number of diagnoses in the program will allow this. To facilitate such objectivemeasurement, methods for efficient data collection regarding tests done in prenatal screeningand diagnostic programs, and the results and outcomes of them should be developed. As I'venoted this will be much easier for some programs than others that are dispersed such asultrasound.ConclusionI've covered a lot of ground, but in summary, most Western countries have reached a levelof health care where congenital and genetic disorders contribute substantially to infantmortality, chronic health problems, and childhood disability. It is possible to prevent somecongenital and genetic diseases if prenatal screening and diagnostic programs are provided.Currently, in developed countries, pregnant women are offered screening and diagnosis for arange of disorders, and this screening is likely to expand in future. Given this, and given the16context that the ability to identify genes is rapidly expanding, the costs and benefits of goingthis route need very careful assessment before we uncritically embrace it. The benefitsinclude informed choice; the birth of healthy children and accepted, affected children; andnormal family life.. These services, if provided appropriately with full information and free choice, are wantedby the public and by most women. But "to ensure ethically and culturally appropriate services,broad input into their planning and evaluation is needed.Experience and data so far indicate that people use the knowledge about the health status ofthe fetus obtained through these programs in a responsible and balanced way. The reason tohave prenatal screening and diagnostic programs is not to save money, and the approach tocost-benefit analysis of such programs should not give the message to the public that it is.The evolving capacities of prenatal screening and diagnosis challenge us as a society tomanage these rapidly changing technologies with wisdom and humanity, I hope we can do it.REFERENCES1. Drummond, M.P. (1980) Principles of Economic Appraisal in Health Care. Oxford UniversityPress.2. Modell, B. and A.M. Kuliev. A Scientific Basis for Cost-Benefit Analysis of Genetics Services.Trends in Genetics, Vol. 9, No. 2, 1993.3. Cuckle, H.S., N. 1. Wald, P.M. Cuckle. Antenatal Diagnosis ofNeuraI Tube Defects in Englandand Wales. In Press.Prenatal Screening Tests• Maternal Serum Testinge.g., Alpha-FetoProtein (MSAFP),Triple-Testing• Routine UltrasoundFigure 1Prenatal Diagnostic Tests• Amniocentesis- _chromosome analysis- AFP and othersubstances- DNA or enzymeanalysis• Chorionic VillusSampling (CVS)• Targeted specializedultrasoundF igure 2Reasons for "Referral to Genetics Centrefor Prenatal DiagnosisDRisk of chromosomal disorderAdvanced maternal age 77.70/0Previous chromosomal 2.40/0Parental chromosomal 0.5%Relative chromosomal 2.3%Chromosomal markerabnormality 0.1%oRisk of"single-gene disorderoRisk of structural anomalyAbnormal MSAFP*Abnormal ultrasound*Previous/family NTDTeratogeno Pregnant woman's anxietyo Other3.6%3.1%2.5%1.4%83.0%1.60AJ10.6%1.3%3.5%* May also indicate increased risk of chromosomal disorder.Type of disorderTrisomies 13, 18 .and 21 (Down Syndrome)Neural Tube DefectsTurner SyndromeXXY,XYY,XXXSyndromes, balanced.translocations, mosaics· Figure 3% womenwhoterminated pregnancy83%76%70% ·30%Figure 4Number of Obstetrical Ultrasound 'ProceduresYear Number of Procedures" Costs1982 - 1983 358,722 $21,174,8941984 - 1985 490,783 $31,871,9711986 - 1987 636,515 $43,748,0191988 - 1989 813,347 $66,618,8511990 - 1991 998,492 $74,649,481* Number of ultrasound procedures associated with obstetrics and gytiaecologyand paid for under the provincial medical care insurance plans for Quebec,Ontario, Menitobe, Saskatchewan, Alberta, and British Columbia. Figures forthe Atlantic provinces are not included, as ultrasound there is paid for underprovincial hospital insurance plans, not medical insurance plans.Figure 5Cost Benefit Analysis of PNDfor Down SyndromeCost of Program• Cost of informationdissemination• Cost of counselling• Cost of amniocentesisprocedure• Cost of laboratorychromosome analysis• Cost of terminationBenefitsAvoiding costs of care for:iii Cardiac and other surgeryII Excess physician andhospital usage. .II Special educationII Residential carefigure 6£60 per at-risk coupleinformed£1.80 per persontested and informedVery small perpersonCost (1987) per benefitfor thalassaemiaINFORMED COUPLE AT RISKPopulation atriskIEducation jd Information --j INFORMED POPULATION I"l" N~;'" --iREASSURANCE I+ve resultIInformation and counsellingIINFO~ CARRIERSInvitoI fer testing-Ir-·N-i-~-?:if-'r-eHREASSURANCE'+ve resultIInformation and counsellingI£670-700per prenataldiagnosis}£1350-1650per homozygotedetected andreplacedBy repeating the cycle, at-risk couplesachieve aHEALTHY FAMILYAverage additional cost £2500Separation (rare)ICHOIil«EUse artificial insemination by donor "c. (rare)No children (only if no prenatal diagnosis),-- _75% HEALTHY,IPREG~AN.Cyl Declined prenatal diagnosis CHILDRENB 1 25% ACCEPTEDFetal sampling - Loss of pregnancy HOMOZYGOTEJ I (few)...J1 Fetal diaj0sis Unaffected ----IHEAL~ CHIUJ I~ Affected (25%).E ICounselling ---No termination of ---I ACCEPTEDI Pregnancy HOMOZYGOTEr'-erm-~in-atl':'-'O-;-O"""'fIPre c .HPRU 88:1RHPRU 88:2RHPRU88:3RHPRU88:4RHPRU88:5RHPRU88:6RHPRU88:7DBarer, M.L. Gafnl, A and Lomas, J.(1989), ·Accommodating RapId Growth In Physician SUpply: Lessons fromIsrael, WarnIngs for Canada·, International Joumal ofHealth Services 19(1):95-115. OrIginally released InFebruBf}'. 1988.Evans, R.G., Barer, M.L, Hertzman, C., Anderson, G.M., Pulclns, I.R. and Lomas, J.(1989), "The LongGoodbye: The Great Transformation ofthe British Columbia HospItal System·, Health Services Researoh24(4):435-459. OrIginally released March. 1988.Evans. R.G. (1989), ·Readlng theMenu With Better Glasses: Aging and Health Policy Research·, In S.J. Lewis(&d.), Aging and Health: Unldng Research and Public Polle,y, lewis Publishers Inc., Chelsea, 145-167. OrIginallyreleased April, 1988.Barer, M.L(1988), ·Regulating Physician Supply: The Evolution ofBrItish Columbia's Bill 41·, Journal ofHealthPolitics, POUe,y and Law 13(1):1-25Anderson, G.M. and Lomas, J.(1989), ·Reglonallzation ofCoronary Artery Bypass SUrgery: Effects on Aooess·,Medical Care 27(3):288-296. OrIginallyreleased May, 1988.Barer, M.L, PUlclns, I.R., Evans, R.G., Heitzman, C., Lomas, J.and Anderson, G.M. (1989), "Trends In Use ofMedical services by theBderly In BrItIsh Columbia·, Canadian M9dlcalAssociation Joumal141 :39-45. OrIginallyreleased July, 1988The Development ofUfl/zatlon Analysis: How. Why, and Where Ifs Going. August 1988. (G.M. Anderson, J.Lomas)D=Discussion Paper R=ReprintHPRU88:8DHPRU88:9DHPRU 88:10RHPRU 88:11RHPRU 88:12RHPRU 88:13RHPRU 88;14RHPRU 89:10HPRU89:2RHPRU89:3RHPRU89:4RHPRU 89:50SquBlfng the Clrole: ReconclUng Fee-tor-Service wfth Global Expenditure Control. September 1988. (R.G.Evans).Practice Patterns ofPhyslclSTl$ wfth Two YBaI'Rssldene,y Versus One YBaI'lntsmshlp Trslnlng: Do Both RoadsLead to Rome? September 1988. (M.T. SChechter, S.B. ~heps, P. Grantham, N. Rnlayson, R. SIzto)Anderson, G.M. and Lomas, J.(1988), "Monitoring the Diffusion ofaTechnology: Coronary Artery BypassSurgery In Ontario", American Journal ofPublic Health 78(3):251-254Evans, R.G. (1988),""We'll Take Care ofItFor You": Health Care In the canadian Community", Daedalus117(4):155-189Barer, M.L., Evans, R.G. and Labelle, R.J. (1988), "Fee Controls as Cost Control: Tales From the Frozen North",The Milbank Qusrterty66(1):1-64 .Evans, R.G. (1900), "Tension, Compression, and Shear: Directions, Stresses and outcomes ofHealth CareCost Contror, Journal ofHealth Polltlcs, Polle,yand Law 15(1):101-128. OrigInally released Decsmber, 1988.Evans, R.G., RobInson, G.C. and Barer, M.L(1988), "Where Have All the ChIldren Gone? Accounting for thePaediatric Hospital Implosion", InR.S. Tonkin andJ.R. Wright (eds.), Redsslgnlng RelationshIps In ChUd Healthcare, B.C. Children's Hospital, 63-76PhysIcian Utilization 8efore andAfterEntering aLong Tenn Care Program: An Application ofM81fcov ModellIng.January 1989. (H. Krueger, AY. Ellencwelg, D. Uyeno, B. McCashln, N. Pagllccla)Hertzman, C., Pulclns, I.R., Barer, M.L, Evans, R.G., Anderson, G.M. and Lomas, J.(1900) "Flat on Your BackorBack toyour Rat? Sources ofIncreased Hospital services Utilization Among the Elderly In British ColumbIa",Social Science and Medicine 30(7):819-828. OrigInally released January, 1989.Buhler, L, Glick, N. and Sheps, S.B. (1988), "Prenatal Care: AComparative Evaluation ofNurse-Mldwlvss andFamily Physicians", Canadian Medica/Association Jouma/139:397-403Anderson, G.M. and Lomas, J.(1989), "Recent Trends In Cesarean section Ratss InOntario", Canadian MedicalAssociation Joumal141 :1049-1053. OrigInally released February 1989.The CanadIan Health care System: AKIng's Fund Interrogatory. March 1989. (A.G. Evans)2HPRU89:6RHPRU89:7RHPRU89:8RHPRU89:9RHPRU 89:100HPRU 89:11RHPRU 89:120HPRU 89:130HPRU 89:14RHPRU90:1RHPRU90:2DHPRU90:3DAnderson, G.M., SpItzer, W.O., WeInsteIn, M.C., Wang, E., Blackburn, J.LandBergman, U. (1990), "Benefits,Risks, andCosts ofPresaiption Drugs: AScIentific Basis for Evaluating Policy Options", alnlcal PharmacologyB1Jd Therapeutics 48(2):111-119. Originally released Aprfl, 1989.Evans, R.G. (1990), "The Dog In the Night l1me: Medical Practice Variations andHeaJth Policy', In T.F.Andersen andG. Mooney (eds.), The Challenges ofMed/caI Practice Variations, The McMillan Press Ltd,London,l17·152. Originally released June 1989.Evans, A.C. (1991), "Ufe and Death, Money and Power: ThePolitics ofHealth Care Rnance", In T.J. Ulman andLS.RobIns (eds.) Health Politics and PoRcy (2nd don) Part 4(15):287-301. Ori~lnally released June, 1989.Barer, M.L, NIcoli, M., Dlesendorf, M. and Harvey, A. (1990), "From Medlbank to Medicare: Trends In AustralIanMedical Care Costs and Use From 1976 to1986", Community Health Studies XIV(l ):8-18. Originally releasedAugust 1989.Cholesterol SC198nlng: Evaluating Alternative Strategies. August 1989. (G. Anderson, S. Brlnkworth, T. Ng)Evans, A.G., Lomas, J., Barer, M.L, Labelle, A.J., Fooks, C., stoddart, G.L, Anderson, G.M., Feeny, D., Gafnl,A., Torrance, G.W. and Tholl, W.G. (1989), "Controlling Health Expenditures -The Canadian ReaJIty", NewEngland Joumal ofMedicine 320(9):571-577The Effect ofAdmission to Long Term Care Program on UtIlization ofHealth Services bythe Elderly InBrItIshColumbia. November 1989. (A.Y. Ellencwelg, A.J. stark, N. Pagllccla, B. McCashln, A. TOUrigny)UtIlization Patterns ofalents Admmed orAssessedbutnotAdmmed to aLong Term care Program ­Characteristics and DIfferences. November 1989. (A.Y. Ellencwelg, N. Pagllccla, B. McCashln, A. Tourigny, A.J.Stark)Anderson, G.M., Pulclns, I.A., Barer, M.L, Evans, A.G. andHertzman, C. (1990), "Acute Care HospitalUtilization Under Canadian National Health Insurance: TheBritish Columbia Experience from 1969 to 1988",InqUiry 27: 352·358. Originally released December, 1989.Anderson, G.M., Newhouse, J.P. andRoos, LL (1989), "HospItal Care for Elderly Patients wIth DIseases oftheCirculatory System. AComparison ofHospital Use In the United states and Canada", New England Joumal ofMedicine 321 :1443-1448Poland: Health and Environment In the Context ofSocioeconomic DeclIne. January 1990. (C. Hertzman)The Appropriate Use ofIntrapartum Electronic Fetal Heart Rate Monitoring. January 1990. (G.M. Anderson, D.J.Allison)3HPRU90:4RHPRU90:5RHPRU90:6DHPRU90:7RHPRU90:8RHPRU 9O:9DHPRU 9O:10RHPRU90:11DHPRU90:12DHPRU90:13RHPRU 9O:14DAnderson, G.M.,Brook, R. and Williams, A. (1991) "A Comparison ofCost-Sharing Versus Free Care InChIldren: Effects on the Demand for Office-Based Medical Care", Med1caJ Care 29(9):890-898. OrIgInallyreleased JanuSI}', 1990. .Anderson, G.M., Brook, R., Williams, A. (1991) "Board certification and Practice style: An Analysis ofOffIce.Based Care", The Journal ofFamIly Practice 33(4):395-400. OrigInally released FebnJBI}', 1990. OrigInallyreleased FebruBl}', 1990.An Assessment ofthe Value ofRoutine Prenatal Ultrasound ScreenIng. February 1990. (G.M. Anderson, D.Allison)Nemetz, P.N., Ballard, D.J., Beard, C.M., ludwig, J.,Tangalos, E.G., Kokmen, E., WeIgel, K.M., Belau, P.G.,Bourne, W.M. and Kurland, LT.(1989) "An Anatomy ofthe Autopsy, Olmsted County, 1935 through 1985", MayoClInic Proceedings 64:1055-1064Nemetz, P.N., Beard, C.M., Ballard, D.J., ludwig, J., Tangalos, E.G., Kokmen, E., Weigel, K.M., Belau, P.G.,Bourne, W.M. andKurland, LT.(1989) "Resurrecting the Autopsy: Benefits andRecommendations·, Mayo alnlcProceedings 64:1065-1076Technology DiffusIon: The Troll Under the Bridge. APilot Study ofLow and HIgh TechnologyInBrfUshColumbIa March 1990. (A. KazanJian, K. Friesen)sapphires In the Mud? The Export Potential ofAmerican Health Care Rnanclng. Enthoven, A.C. (1989), "WhatCan Europeans Learn from Americans?·, Evans, R.G., Barer, M.L. (1989), Comment Health care FinancingReview, Annual Supplement 1989Healthy Community IndIcators: The PerfIsof the S9arch and the Paucity ofthe Find. March 1990. (M. Hayes, S.Manson Willms)Use ofHMRI Data InMneteen Brftfsh Columbia Hospitals and Future Directions for case Mix Groups. April1990. (KM. Antioch)Evans, R.G. and Stoddart G.L(1990) ·Produclng Health, Consuming Health Care·, Social Science and Med1c1ne31 (12) 1347·1363. OrigInally released Aprfl, 1990.Automated Blood Sampl~Handllng Inthe ClInIcal Laboratof}'. June 1990. fN. Godolphln, K. Bodtker, D. Uyeno,L-Q. Goh)4HPAU9O:15AHPAU 90:160HPAU9O:170HPAU 9O:18AHPAU 90:190HPAU9O:200HPAU9O:21DHPAU9O:22AHPAU9O:230HPAU 90:240HPAU 91:1RHPAU91:2DHPAU91:3AHPRU91:4D-Anderson, G., Sheps, S.B.,CardIn,K, (1990) "Hospital-based UtillzaUon Management aCross-canadaSurvey", CanadIan MedTcalAssoclstlon Journal 143 (10):1025-1030. Originally released June, 1990.HospltaJ.BasBd Utlflzation Management ALltBrature Review. June 1990. (S. Sheps, G.M. Anderson, K Cardiff)Renections on the Rnancfng ofHospital Capital:A Canadian Perspective. June 1990. (M.L Barer, A.G. Evans)Evans, R.G. Barer, M.Land Heitzman, C. (1991), "The 2O-Year Experlment Accounting For, Explaining,andEvaluating Health Care Cost Containment In canada and the United states",Annual Review ofPublic Hsalth12:481-518. Originally reieas8d September, 1990.Accssslbie, AcceptablB andAffordable: Rnanclng Haalth Care In canada september 1990. (A.G. Evans)Hungary Report OCtober 1990. (C. Hertzman)Unavailable for Circulation.Anderson, G.M., Pulclns, I. (1991), "Aecenl trends In acute care hospllal utilization In Ontario for diseases oftiecirculatory system", CMAJ 145(3):221-226. Originally released October, 1990.Environment andHealth In Czechoslovakia December 1990. (C. Hertzman)Perceptions and RsaJJtles: MBdical andSurgical Procedure Variation AUterature Review. January 1991. (5.Sheps, S. SCrivens, J. Galt)Nemetz, P.N.,Tangalos, E.G.and Kurland, LT. (1990), "The Autopsy and epidemiology - Olmsted County,MInnesota and Malmo, Sweden", APMIS98:76S-785PuttIng Up orShutting Up: Int8lpf9ting Health status Indicators From An Inequltles perspsctiVe. May 1991. (C.Hertzman, M. Hayes)Barer, M.L (1991), "ControJnng Medical Care Costs In canada" (EdItorial), JoumsJ ofthBAm8rlcan M8dIcaIAssociation 265(18):2393-2394The M99ting ofthe Twain: Managing Health GarB Capital, CapacityandCOsts Incanada. June 1991. (M.LBarer, A.G. Evans)5HPRU91:5RHPRU 91:60HPRU 91:70HPRU 91:80HPRU 91:90HPRU 91:100HPRU 92:1RHPRU92:2RHPRU92:30HPRU92:4RHPRU92:50HPRU 92:60HPRU 92:70-Barer, M.L, Welch, W.P. and AnUoch, L (1991) ·Canadlan-Amerlcan Health Care Comparisons: ReftecUons OnThe HIM'S Analysis·, Health Affairs 10(3):229-239. Originally released June, 1991.Toward Integrated Medical Resource Policies for canada: Background Document June, 1991. (M.L Barer,G.LStoddart) Cost: $45.00Toward Integrated Medical Resource Policies for Canada: Appendices. June, 1991. (M.L Barer, G.LStoddart)Cost: $30.00Bulgaria: The Public Health Impact ofEnvironmental Pollution. Augus~ 1991. (C. Hertzman)Renections on the Revolution In Sweden. September, 1991. (R.G. Evans)The Canadian Health Care System: Where are We; How Old We Get Here? OCtober, 1991. (R.G. Evans, M.M.Law)Barer, M.L., Hertzman, C., Miller, R., Pascali, M.V. (1992) ·On Being Old and Sick: The Burden ofHealth Carefor the 8derly In Canada and the United states·, JoumaJ ofHealth Politics, Polity and LawVol.17(4)763-682.Originally released December, 1991. No charge.Manson Willms, S. (1992) ·Houslng for Persons with HIV Infection In Canada: Health, Culture and Context",Westem Geographic series Vol. 26:1·22. No chargeEnvironment and Health In the Baltic Countries. April, 1992. (C. Hertzman) Cost $8.00Barer, M.L, Evans, R.G. (1992) ·lnterpreUng Canada: Models, Mind-Sets, and Myths·, HeaJthAffalrs 11(1):44-61No chargeAids RIsk Taking BehaviourAmong Homosexual Men: SocIo-demographlc Markers and Polity ImpUcations.June,1992. (R.S. Hogg, K.J.P. Craib, B. Willoughby, P. Sestak, J.S.G. Montanar, M.T. Schechter)Cost $5.00The AdequatyofPrenatal Cars and Incidence ofLow BIr1h WeightAmong thePoor In Washington State andBrftlsh Columbia. June, 1992. (S. Katz, R.W. Armstrong, J.P. LoGerfo) Cost $5.00Medication Profiles on Admission vs. DIscharge In Patients ataGeriatric Referral Centre. November, 1992.(J.H. Schechter, M. Donnelly, M.T. SChechter) Cost $8.006HPRU 92:80HPRU 92:90HPRU 92:10RHPRU 93:10HPRU93:2RHPRU93:3RHPRU93:4RHPRU93:5DHPRU93:6DHPRU93:7DHPRU93:8RHPRU93:9DHPRU93:10R-What seems to be the Problem? The InternationalMovement to Restructure Health Care Systems. November,1992 (A.G. Evans) $8.00A CompendIum ofStudIes on Environmental RIsk Perception In B.C. November, 1992. (C. Hertzman, S. Kelly,A.S. Ostry, D. SClarretta. KKKTeschke)Cost $8.00Anderson, J.M., Blue, C. Lau, A. (1992) "Women's Perspectives on ChronIc Illness: Ethnlclty, IdeologyandRestructuring ofute", Soc.SCI.Med. Vol.33 12, pp 101-113. No charge .Evaluation ofVI-Care: A UtIlization Management Program ofthe Greater Victoria Hospital Society. January.1993. (G. Anderson, S. Sheps, K Cardiff) Cost $12.00Barer, M.L, Stoddart, G.L(1993) "Toward Integrated Medical Resource PoDcles for Canada: canadian MedicalAssociation Jouma/ Series", canadian MedIcalAssocIation Jouma/146 (3). (5), (7), (9). (11); 147 (1), (3), (5), (7),(9), (11); 148 (1) $12.00Evans, A.G. (1993) "The Canadian Health-Care Rnanclng and Delivery System: Its Experience and Lessons forOther Nations·, Yale Law &PoRcy Review 10(2) No chargeTan, J.KH., McCormick, E., Sheps, S.B. (1993) ·Utllization Care Plans and Effective Patient Data Managemenr,Hospital &Health Services AdminIstration 38:1 Spring No chargeNursing Resources In Brlt/sh ColumbIa: Trends, Tensions and Tentative Solutions, February, 1993. (A. KazanJian,L Wood) Cost $8.00Hospital Rnanclng In Canada April, 1993. (M.L Barer) Cost $8.00OUtcomes Managementand Resource Allocation: How should Qualityofutebe Measured? July, 1993 (D.C.Hadom) Cost $8.00. .Evans, A.G.(1993) ·HeaIth Care Reform: The Issue From Hell" Ponc.y Options 35-41 No. chargeUser Fees for Health Care: Why aBad Idea Keeps ComIng Baclc. August, 1993 R.G. Evans, M.LBarer, G.LStoddart cost $8.00Anderson, G.M. Keriuke, KJ. Pulclns I.A. Hertzman, C. Barer, M.L "Trends and Determinants ofPrsscrfpUonDrug ~nd/tures In the Elderly: Data from the British Columbia Pharmacars Program" Inquiry 30: 199-207SUmmer 199~ No charge7HPRU 93:11RHPRU 93:12DHPRU 93:13DHPRU 93:14DHPRU 93:15DHPRU 93:16DHPRU 93:17DHPRU94:1D. HPRU 94:1RHPRU 94:2RHPRU94:3D- -Hertzman, C. -Environment&Health: InCentral and Eastern Europe - Q>st $12.00Why Not User Charges? The Real Issues. December 1993. (G.L Stoddart, M. L Barer, R. G. Evans, V. Bhalla)cost $8.00Who Are the Zombie Masters, and What Do They Want? December 1993. (R.G. Evans, M. L Barer, G.LStoddart, V.Bhatia) cost $5.00User Charges, Snares and Delusions: Another Look atthe Uterature. December 1993. (G. L Stoddart, M.LBarer, R.G. Evans,) cost $8.00The Remarkable Tenacity ofUser Charges: AConcise History ofParticipation, Positions, and Rationales ofcanadian Interest Groups In the Debate over "Direct Patient Participation"ln Health Care Ananclng. December1993 (M.L Barer, V. Bhatia, G.L Stoddart, R.G. EVans,)cost $8.00IrsNot the Money, Irsthe Principle: Why User Charges for Some Services, and Not Others. December . ( R.G.Evans, M. L Barer, G.LStoddart, V. Bhatia) December 1993 Cost $8.00Charging Peter toPay Paul: Accounting for the AnanclaJ Effects ofUser Charges. December 1993 ( R.G. Evans,M. L Barer, G.LStoddart) Cost $12.00Tip-toeIng Toward Health Care Reform In the United states. January 1994. (M. L Barer, E. Morrison, I.Morrison) Cost $5.00Barer, M.L, Morrison, E., Morrison, I.(1994) "canadian Physicians may hear footsteps ofchange asUS tiptoestoward health care reform", canadian MedicalAssociation Journal, Vol 150 (6), P980-987.This Is arevfsed and shortened version of94:1DEvans, Robert G. "Less IsMore: Contrasting Styfes In Health CarB' Canada and the United states, Differencesthat Coun~ 21 -41. No chargeAvalanche orGlacier. March 1994. ( M.LBarer, R.G. Evans, C. Hertzman) Cost $8.008

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