1 Degree project, 30 ECTs January 8, 2019
Abdominal Aortic Aneurysm Screening
an Ethical Discussion
Version 2
Author: Ami Holmström, MB
School of Medical Sciences Örebro University Örebro Sweden
Supervisor: Rolf Ahlzén, Associate Professor
Faculty of Humanities and Social Sciences Karlstad University Karlstad Sweden Word count Abstract: 216 Manuscript: 6982
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Abstract
Introduction: Abdominal aortic aneurysms (AAA) have a prevalence of approximately 2%,
and are more common in men. AAAs are generally asymptomatic, but if ruptured and
untreated, the mortality rate is close to 100%. Screening programs for AAAs are implemented in Sweden, the UK, and the US. This study describes the different views of AAA screening with a special emphasis on underlying ethical issues.
Aim: To analyze the scientific background of AAA screening in order to be able to discuss its
ethical basis.
Methods: This was a qualitative literature study with an analysis of arguments using a
hermeneutic method. Articles were obtained through a literature search and consisted of official articles, scientific articles, and debate articles.
Results: A recent dissertation has questioned the value of AAA screening because of
decreased AAA mortality and risk for overdiagnosis. However, most studies and official recommendations are in favor of AAA screening because disease specific mortality decreases and the screening program is considered cost-effective.
Conclusion: This study shows that intellectual passion has created an unusually polarized
discussion. It seems that benefit outweighs harm. Since AAA screening is the first screening program which could lead to the death of a previously asymptomatic individual, well founded informed consent is extremely important. Finally, both decisions to act and not to act have moral consequences.
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Introduction
Abdominal aortic aneurysm
An aneurysm is a weakening of the blood vessel wall, leading to a localized dilatation of the vessel including all the layers of the wall [1]. An abdominal aortic aneurysm (AAA) is a dilatation of the abdominal aortic diameter to more than 3.0 cm, or a diameter of 1.5 times the normal aorta. The most common site for AAA is infrarenal [2]. Risk factors of importance are age over 65 years, male sex, and smoking. Coronary artery disease, cerebrovascular disease, hypertension, atherosclerosis, and hypercholesterolemia are also associated with a higher risk of developing AAA. Growth rates of AAA vary depending on the diameter, with larger aneurysms growing faster [3]. The risk for rupture of an AAA is related to its size. The yearly risk for rupture for AAAs with a diameter of 3.0 to 3.9 cm is close to 0%, for those 4.0 to 4.9 cm 1%, and for those 5.0 to 5.9 cm 11% [4].
Prevalence of AAA varies greatly between regions because of different definitions, distribution of risk factors, age, and gender of study groups. It is 2.2% in the US, 2.5% in Europe, 6.7% in Australia, and 0.5% in Asia. Men are four to six times more affected than women. The prevalence in 55-64 year olds is 1.3%, 2.8% in 65-74 year olds, 1.2% for 75-84 year olds, and 0.2% at an age over 85 years [5]. The majority of AAAs are asymptomatic and are often detected en passant. If ruptured, symptoms are acute abdominal or back pain, hypotension and shock. 59 to 83% of patients die before they reach a hospital or undergo surgery. If untreated, the mortality rate of ruptured AAA is close to 100% [2]. In 2014, the Swedish Cause of Death Register identified 506 deaths as caused by AAA. Due to the autopsy rate in Sweden being only 7%, these numbers are probably an underestimate [6].
Screening
Screening is a systematic examination of a part of the population made to identify persons with a condition that implies a high risk for future disease [7]. The purpose is to detect conditions that may lead to severe or extensive consequences for the individual such as premature death, severe injury or disability, but also for society a large consumption of resources. The goal is early intervention for the condition and thus to decrease the consequences for the population [7].
The Swedish guidelines for screening are inspired by a public health paper published by WHO in 1968 in which Jungner and Wilson outlined principles for screening: The condition being screened for should be an important health problem with a well-known natural history
4 and a detectable early stage. There should also be a suitable test method, early treatment should provide more benefit than discovery from clinical symptoms, and the screening program should decrease mortality, morbidity or loss of function associated with the
condition. Both the test method and treatment should be accepted by the population and health benefits should be larger than negative effects. Finally, the screening program should be ethically acceptable and cost-effective [8,9]. (For a translation of these criteria, see appendix). In 2018, The National Board of Health and Welfare in Sweden recommends screening
programs for the following subpopulations: ultrasonography for AAA in men aged 65 years, mammography screening for breast cancer in women aged 40 to 74 years, screening with cytology and HPV testing for cervical cancer for women aged 23 to 64 years, fecal
hemoglobin test for colon and rectal cancer for women and men aged 60 to 74 years, and a selection of congenital diseases for newborns [10].
The current screening programs for AAA are based on the data from four large randomized controlled trials performed in the UK, Denmark and Western Australia during the eighties and nineties, including over 130,000 participants [11–14]. The US Preventive Services Task Force reviewed these randomized controlled trials along with other studies on AAA screening in 2005, and concluded that screening reduced AAA-related mortality [15].
Screening programs for AAA are currently implemented nationally in Sweden, the UK, and the US. Several other countries (among them The Netherlands, Italy, Denmark, Norway, and Australia) have ongoing or have previously had randomized controlled trials for a screening program but not on a national level. A one-time screening is recommended for all men at the age of 65 in Sweden and the UK, and the US recommends screening for men between 65 and 75. The US only screens persons who have smoked more than 100 cigarettes in their lifetime, and also offers screening for women with a family history of AAA [16]. In Sweden, 75-85% of all invited men participate in the screening program, which is comparable with other
national screening programs [9]. The preferred method for screening is using ultrasonography, which is uncomplicated, harmless, and inexpensive and has a sensitivity of 95% and a
specificity approaching 100% [2]. Follow-up is recommended when the diameter of the abdominal aorta is wider than 30 mm, and a diameter over 55 mm is an indication for elective surgery [16].
5 In spite of these official recommendations there are those who are not convinced about the merits of AAA screening. This paper describes the different views with a special emphasis on underlying ethical issues.
Principles of biomedical ethics
The traditional ethical standard for doctors is the Hippocratic oath which was named after the Greek physician Hippocrates, circa 460–370 BC, and is still used in a modified form in many countries. It states that “I will use treatment to help the sick according to my ability and judgment, but never with a view to injury and wrong-doing”. The phrase ‘do no harm’ (primum non nocere) is often attributed to Hippocrates but actually dates from the 17th
century [17]. Today the Hippocratic oath is believed to be too paternalistic and has largely been replaced by other principles, some of which are mentioned below.
There are four well-known and very influential principles of biomedical ethics. They are not considered absolutes, but provide powerful action guides in the clinical setting, e.g. in the discussion of treatment options. The principle of respect for autonomy, which implies that patients have the right to make their own choices after receiving accurate information, is the basis of informed consent. The principle of non-maleficence requires that health care
professionals do not intentionally cause injury or harm to the patient. The principle of
beneficence implies that health care providers have a responsibility to benefit the patient, and also to prevent and to eliminate harm from the patient. The principle of justice involves reasonable distribution of services in the society. These principles used to be non-hierarchical, but autonomy is now considered the most important. Sometimes the principles can be in conflict with each other. Health care professionals have a prima facie duty, that is, a duty to uphold all of the principles. However, in the actual situation, the potential risks and benefits of the principles need to be balanced and the principle that is most significant should be determined [18].
Key concepts
There are some recurring concepts in this paper that will facilitate reading if explained first. ‘Numbers needed to screen’ indicates the amount of people that need to undergo screening during a given time period to save one life or prevent one adverse event [19]. ‘Overdiagnosis’ is when an asymptomatic individual is diagnosed with a condition that, if not found because of testing, would not have caused any symptoms or death during the individual’s lifetime. ‘Medicalization’ is when non-medical features become considered medical issues in an
6 individual’s life [20]. ‘Overtreatment’ is treatment of a condition that would have disappeared on its own or never given any symptoms [21]. ‘Cost-effectiveness’ is a way of allocating limited health care resources by comparing costs and effects of an intervention [22].
Aim
The aim is to analyze the scientific background of AAA screening in order to be able to discuss its ethical basis. This was done by answering the following research questions:
1. What is the current scientific background concerning AAA screening? 2. How can ethical principles facilitate evaluation of harm and benefit of AAA
screening?
Methods
This is a qualitative literature study, with an analysis of arguments. The data collected
included debate comments, books, and webpages. When searching for articles, the aim was to find as broad a spectrum of articles covering the subjects of AAAs, screening, and ethics, as possible. Online search for scientific articles was made mainly by using PubMed and Google Scholar, and included articles ranging from large studies of international, wide covering journals, to debate comments from smaller national journals. Terms used for searching were “abdominal aortic aneurysm”, “screening”, “ethics”, “autonomy”, “informed consent”, “integrity”, “medicalization”, “overdiagnosis”, “overtreatment”, “quality of life”,
“psychological effects”. These terms were combined in different constellations, most of which contained “abdominal aortic aneurysm”. A search for articles written by M Johansson, who will be introduced later in this paper, was also made. The search was limited to texts available in full text and written in English or Swedish. It was difficult to find articles which covered the ethical aspects of AAA screening, and a specific systematic search strategy was not conducted. Instead, the focus for the results was to include large randomized control trials by searching for the terms mentioned earlier, and official recommendations about AAA
screening. As for the discussion, articles were included if they contained more than a few sentences about ethical considerations. In total, 40 articles found on PubMed and Google Scholar were used. Five articles were obtained from the reference list of the articles found. To look into the debate, three debate comments were located on the website of a journal
published by the Swedish Medical Association by searching for articles written about AAA screening in the year of 2018. To study the Swedish guidelines for screening, three references
7 were found on The National Board of Health and Welfare, and two on Swedish Agency for Health Technology Assessment and Assessment of Social Services.
The texts were analyzed using a hermeneutic method with text interpretation as the main focus. The aim of using a hermeneutic interpretation method is to find a valid and mutual understanding in the meaning of a text [23]. This can be described as a spiral, where
interpretation arises and leads to a preunderstanding for the upcoming understandings. From this point the reader cannot return to the previous point of understanding. The understanding is constantly deepened, and the interpretation is in essence endless. The author’s personal pre-understandings and preconceptions of the subject would inevitably be an interference in the analysis of the texts, and efforts to put these pre-understandings aside were made, but there was awareness of the fact that it was not possible to completely do so. Furthermore, the author did not have the ability either to make a fully neutral interpretation of the texts, or to come to a conclusion of an objective truth.
Epidemiological statistics were included in the articles analyzed, however no additional statistics were made in this study, due to the study design.
Results
Studies in favor of and recommendations for AAA screening
Among the four randomized controlled studies that made up the base for the screening
programs of today, the Multicentre Aneurysm Screening Study (MASS) performed in the UK between 1997 and 1999 was the largest, including 67,800 participating men aged 65 to 74 years. Of the 33,389 screened men, 4.9% had AAA (n=1333). The screened men underwent elective surgery three times more often than men in the control group, but the non-screened men underwent emergency surgery twice as often. Sixty-five men in the screened group died because of AAA, compared to 113 in the control group, which resulted in a 42% risk
reduction for the screened group. The incidence of non-fatal ruptured AAAs was lower in the screened group. No significant difference in all-cause mortality was found between the groups. Sixteen men in the screening group died after undergoing elective surgery. Moreover, 66 men in the same group suffered a ruptured AAA. Thirty-eight of these patients were not available to benefit from treatment to prevent the rupture, since they did not fulfill with the screening and intervention program or were unfit for surgery. Measures for anxiety,
8 both groups, and showed no difference between men with negative and positive screenings. The study concluded that screening would be beneficial [13].
In the 13-year follow-up of the MASS, the relative risk reduction1 in the screened group compared to the control group was 42%. For each 10,000 men screened, 75 ruptured AAAs were prevented. Non-fatal AAA ruptures occurred twice as often in the non-screened group over the follow-up time. Deaths not related to AAAs were similar between the groups, but a 3% reduction in all-cause mortality was seen in the screened group. The reduction in AAA-related mortality was estimated to 52% in the screened group. The mortality benefit was slightly less 10-13 years after screening, partly due to ruptured AAAs in men with normal findings. After a normal first scan, 59 ruptured AAAs occurred, of which 80% were fatal. Screening was considered cost-effective [24].
It is worth noting that widespread repair of AAAs smaller than 5.5 cm may threaten the cost-effectiveness established in the major AAA trials and could even lead to “more procedural deaths than rupture deaths prevented” [25].
In a 2016 systematic review, the US Preventive Services Task Force updated their 2005 recommendation statement. They recommend a one-time ultrasonography screening for AAA in men of the age 65 to 75 years who have smoked more than 100 cigarettes in their lifetime, as AAAs are most prevalent in this population [4]. The review caused some confusion because the original report did not show a reduction of total mortality because of incorrectly “rounded values rather than P-values” [26]. However, the final report, which included combined data did show that screening decreases total mortality.
The Swedish National Board of Health and Welfare recommends in their 2016 report a one-time ultrasonography screening for AAA for 65 year-old men [9]. This recommendation is based on a review of 135 studies and articles. The comprehensive assessment is that the screening program results in health benefits that outweigh the negative effects. Screening for AAA reduces the mortality in ruptured AAA with about 50% in 65 year-old men, which is the determinant for the recommendation. Furthermore, 84% of all ruptures occur in men older than 65 years. The screening is estimated to prevent premature death in 90 to 100 men per year in Sweden. To prevent one death caused by ruptured AAA, 500 men need to be screened. Early detection of AAA provides an opportunity for elective, preventive surgery, which leads
1 Relative risk reduction = the reduction in the risk of AAA mortality in the screened group versus the control group
9 to a lower risk for the patient, regarding both mortality and severe complications. The
evaluation of the screening program is based on the National Board of Health and Welfare’s screening model, which shows that 12 out of 15 criteria are fulfilled, and that the remaining three are partly fulfilled. The partly fulfilled criteria are related to the information given about the program, organizational aspects of an equivalent screening program, and a plan for
assessment of the screening program’s effects. It was concluded that there is a need to develop and improve these areas. The AAA screening program is implemented and considered effective in all counties in Sweden.
In the 2017 Danish five-year follow-up of the Viborg Vascular (VIVA) trial [27], a randomized control trial, the cost-effectiveness of screening for AAA, peripheral artery disease, and hypertension was studied. The absolute risk reduction (ARR) in overall mortality for the screened group compared to the control groups was 0.0062, and the number needed to invite was 169. There was a significantly larger proportion of men in the screened group who were prescribed prophylactic medication and underwent elective surgery. The total healthcare cost was EUR 148 more in the screened group after five years of follow-up, caused by slightly higher use of service for prescription medication, primary care, and hospital service. The ICER (incremental cost-effectiveness ratio), defined as the cost to gain an extra life year or QALY (quality adjusted life year, a quantification of the health effect by multiplying years of life by health status [28]) was EUR 6872 and EUR 2148 respectively. The probability of screening being cost-effective was 98% and 99% at a willingness-to-pay threshold of EUR 40,000. The authors concluded that vascular screening was cost-effective [27]. According to Swedish guidelines, a low cost is defined as below EUR 10,000, and a high cost is above EUR 50,000 [29]. The threshold for cost-effectiveness in England and Holland is EUR 20,000-30,000, and for the US it is EUR 45,000-90,000.
In 2017 Oliver-Williams et al. [30] published a study with findings from a 25-year screening program which analyzed prevalence and growth rates of small and medium AAAs. They found a “dramatic” 12% reduction of mean aortic diameter from 2.0 to 1.7 cm, but the growth rate of small aneurysms with a diameter of 2.6-2.9 cm remained unchanged. More than 50% developed an AAA of 3.0 cm within 5 years. At a mean age of 76 years 30% had developed a large AAA. A re-screening at age 70 was suggested.
2 ARR = the study showed 10.8% deaths in the control group, and 10.2% in the screened group, which is a reduction of 0.6 percentage points
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In 2018 the Canadian Task Force on Preventive Health Care pooled results from AAA screening from the MASS, Chichester, Viborg, and Western Australia, and found a relative risk reduction for AAA mortality of 43% after a short term follow up of 3-5 years [28]. All-cause mortality was not reduced after a short term follow up, but after 13-15 years there was a significant effect. By the age of 80 years one in 311 men had his life saved. It was estimated that 100,000 Canadians could have their lives saved by the implementation of AAA
screening.
Critique against AAA screening and surgical complications
A doctoral dissertation by Johansson et al. published 2018 in The Lancet [31] was a registry-based cohort study, with the aim to estimate the effects of the AAA screening program in Sweden, observing the mortality from AAA, incidence and overdiagnosis, AAA surgery rates, and overtreatment. Data were collected from men who participated in the screening program, and compared with age-matched men not attending screening. The results showed that AAA mortality decreased with over 70% in both screened and non-screened populations during the period of 2000 to 2015. The follow-up time for the study was six years after screening, which resulted in a decrease of the AAA mortality with 30% in the screening population compared to the control population, equivalent to two men avoiding death because of AAA per 10,000 men recommended screening. The screening resulted in 49 possibly overdiagnosed men and 22 extra elective surgeries per 10,000 screened men. 63% of the additional elective surgeries in the screening population might have been equivalent to overtreatment.
Based on these findings, the authors argued that the decrease in AAA mortality probably was a result of factors other than screening, most importantly reduced smoking rates. Many AAAs could also be detected incidentally because of the increased use of radiologic scans, implying that even in the absence of a screening program, a large part of AAA are found before rupture. Johansson et al. discussed the harms of surgery for AAA in a 2015 article in BMJ. Elective surgery for AAA can be followed by complications such as impotence, myocardial infarction, respiratory failure, renal failure, and prosthetic graft infections [32]. Stather et al. found in a 2013 BJS systematic review [33] that patients who underwent endovascular aortic repair (EVAR) had a significantly lower 30-day mortality rate (1.3%), compared to patients who underwent open repair (4.7%). However, after two years there was no significant difference in all-cause mortality between the two interventions (14.3% versus 15.2%), and this was
11 sustained after four years (34.7% versus 33.8%). Furthermore, a significantly larger fraction of patients who underwent EVAR needed re-intervention or suffered from ruptured AAA.
Debate after Johansson’s dissertation
In a 2018 debate article in a Swedish medical journal, the Swedish Aneurysm Screening Study Group (SASS) responded to Johansson who wrote the doctoral dissertation published in The Lancet [31], and pointed out what they saw as inaccuracies of the paper, stating it was misleading [34]. By referring to a randomized trial made in Sweden from 2016 [35], the authors claimed that the AAA related mortality rate had been reduced with 4% per year among elderly men since the implementation of the screening program. Furthermore, the number needed to undergo surgery to prevent one death caused by AAA rupture, was two. Six-hundred sixty-seven men needed to be screened to prevent one death from AAA. The study concluded that the screening program is still effective in today’s low prevalence in Sweden. Since the introduction of the screening program, 1,500 men have undergone
preventive surgery and therefore the lives of 750 men have been extended by a mean of eight years, and ruptures have become a much rarer diagnosis. A substantial general survival benefit that is not aneurysm related was also found, possibly because patients with newly detected aneurysms stop smoking, adopt a healthier lifestyle, and receive secondary
prophylactics, which contribute to the health benefit of screening. The authors of the debate comment noted that Johansson had limited the follow-up time of screening to six years, although the maximum effect occurs after fifteen years, leading to underestimated benefits of screening. They also pointed out that the study did not register men whose AAAs were
identified by screening, meaning the prevalence was only a third of the actual prevalence [34]. In response to this comment, Johansson stated that the follow-up time was not too short, referring to the results of the MASS study that showed a 50% relative reduction of deaths after four years, and that the absolute effect on death caused by AAA would decrease with 93% with the same follow-up time as in the MASS. If the same trend of the MASS was applied to Sweden, the result would be three instead of two less deaths per 10,000 men after 13 years. The absolute risk reduction was 0.02% calculated in the study. The authors argued that the method used by the SASS group overestimated the effects of screening because of the simultaneous large reduction in AAA caused deaths, regardless of screening. The lower prevalence shown in the study was caused by the study group not receiving correct data on the starting dates of the screening programs from different counties. Additionally, the argument that screening would reduce the total mortality through lifestyle changes seemed unlikely, and
12 reference was made to the US Preventive Services Task Force evaluation in a report from 2014 that screening did not reduce total mortality. The authors suggested a new, independent review of the screening program in Sweden [36].
As a last reply in the debate, the SASS group pointed out that the US Preventive Services Task Force later on published a correction where they concluded that screening does reduce total mortality, meaning Johansson used selective citation. They also commented that an independent review of the screening program advocated by Johansson has already been made by The National Board of Health and Welfare and The Swedish Agency for Health
Technology Assessment and Assessment of Social Services in 2016 [37].
Discussion
Benefits and harms of AAA screening need to be carefully identified and balanced. The fact that it is the first screening program that has the potential risk to result in the death of
otherwise asymptomatic persons through surgical intervention [38] requires evaluations based on a solid scientific and ethically acceptable foundation. The purpose of the following
discussion is to illustrate how common ethical principles can facilitate the interpretation of the known relevant scientific facts about AAA screening, but some initial comments about how researchers reach contradictory conclusions seem to be in order.
Polarization of scientific communities
The debate about AAA screening raises an inevitable suspicion about strong personal conviction and prestige behind a sometimes thinly disguised veil of scientific presentation. Groups of researchers publish research which support their previous findings year after year. The risk of biased research is increased even if the researchers have an honest conviction about being right and do not identify themselves as being members of a polarized group. The International Committee of Medical Journal Ethics has identified ‘intellectual passion’ as a source of potential bias. Polarization could be individual passion but has also been found to be, at least partly, a group phenomenon [39].
Ploug et al. analyzed two articles by Danish authors about AAA screening which were published in BMJ in 2009 and 2012 [40]. The first study found a less than 30% chance of screening being cost effective at a threshold of GBP 30,000. The second paper found a 92% probability of screening being cost effective at a threshold of GBP 20,000. It was found that both studies based their estimates from sources which would favor the conclusion of the study. It was also found unlikely that the choices of values were based on unconscious
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processes because of their ‘systematic duration and extent’. The differences were regarded as symptomatic of conflicts between distinctive scientific groups about screening programs in general. The papers were regarded more as interventions in a political debate than scientific research.
An analysis of Johansson’s dissertation and other papers from this perspective may serve as an interesting example of research polarization. Johansson’s dissertation consisted of four papers. The first two were a Cochrane systematic review which concluded that benefits and harms remain uncertain for screening of malignant melanoma which should only be
performed as randomized trials. Papers three and four questioned the continued use of AAA screening. Since 2015 Johansson has written numerous articles and comments about how screening causes harm, overdiagnosis, psychosocial risk, and how informed consent permits overmedicalization. According to Johansson emotional and cultural influences might affect a choice more than intellectual information, the non-screened individual might feel guilty if they later on develop the condition, information is harmful if it leads to unjustified stress or interventions that could harm, no medical practitioner is fully neutral, and time and resources should not be focused on healthy persons but on those who are already ill [41].
Johansson is a member of Cochrane Sweden and part of her dissertation has been co-written with Peter Gøtzsche from Nordic Cochrane Center. Gøtzsche has written extensively about mammography and cancer screening which he regards as harmful and thinks should be abolished. In 2018 he was kicked out from Nordic Cochrane Center after publishing a report which claimed that a Cochrane Collaboration documentation about HPV vaccine was
incomplete and biased [42]. The report was refuted by Cochrane International in a 30 page evaluation. Nordic Cochrane Center was described as an organization which does not seek scientific clarity but rather fights for certain ideas which makes it a political actor. It is impossible to know how much Gøtzsche influenced Johansson’s work, but he is a highly controversial figure far from mainstream medicine which perhaps could raise some doubts about both his and her objectivity.
Johansson and the researchers around her display a pattern of distrust of intervention directed screening which does not involve symptomatic patients and their evaluation of benefit versus harm differs from that of other researchers. They have argued that screening always causes harm, fails to catch disease early and that the tendency of screened groups to be treated with more drugs increases mortality. All of these claims have been repudiated by several other researchers. Gøtzsche also firmly believes that the primary outcome should not be
disease-14
specific but total mortality [43]. Critics of Gøtzsche have pointed out that total mortality is a legitimate end point in treatment studies in which all patients have the disease and most would die from it, but looking for the same end point in screening trials in which most participants die for other reasons “shows a lack of understanding”. Studies which show an effect on total mortality may require hundreds of thousands of participants, or even millions, and follow-up periods of ten to 20 years which would make them unrealistically large and impossible to perform [44].
Beneficence versus non-maleficence
In order for a screening process to be considered ethical it must be shown that it leads to more benefit than harm for the screened population. Our present knowledge of the balance between benefit and harm have been outlined in detail in the results section. The relatively slow progress of AAA, the well-known natural history and the reliable diagnostic process fulfill many criteria for successful screening. The above studies (MASS, VIVA, SASS) and the recommendations of the US Preventive Services Task Force show that screening for AAA could reduce and prevent deaths from sudden ruptures by about 50%. It is therefore generally believed, by researchers, government organizations, and ethical committees, that AAA screening benefits the groups of 65-year-old men which are screened. There are some who would disagree with this position since there is a risk that a previously asymptomatic individual will have AAA diagnosed and die from perioperative complications. It can be argued that this violates the rights of an otherwise healthy person [38].
AAA screening illustrates a conflict between the principles of beneficence and
non-maleficence. One may question, as Johansson does, if improving the prognosis of some while causing others harm, e.g. perioperative morbidity or psychosocial harm, is ethically
acceptable. Is there any way to justify the bad luck of the asymptomatic man who dies on the AAA operating table? An attempted answer is related to the biomedical principle which many regard as the most important.
Autonomy
Autonomy means that adult competent persons have a right to make their own choices and live according to their wishes as long as they do not hurt others. In health care autonomy means that the principle of informed consent is of the utmost importance. The patient must be informed of different treatment options and his/her opinions should be respected even if they differ from what the care giver believes is the most rational solution.
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Informed consent is regarded the key to patient autonomy and should, according to O’Neill, provide assurance that the patient is neither deceived nor coerced. On the surface informed consent might seem easy and straightforward but in reality it is more complicated and remains utopic according to Johansson [45]. It has been shown that evaluations of risky situations are often dominated by emotions rather than logic [46]. Irrational decisions are also often made because even well-educated adults have difficulties understanding numbers, especially low probabilities [47].
It may also be argued that the real danger is not a lack of informed consent, but that the decision about whether to accept screening or not is not entirely free since the invitation to AAA screening is a government approved initiative which implies some kind of responsibility not to reject it [38].
Another concern is nudging, which could be defined as a subtle way of influencing decisions or behavior in a predictable way without forbidding any options. Smoke free areas and prohibition of tobacco advertising are examples of nudging. The object of nudging in
screening is to increase attendance and promote ‘the healthy option’ [40]. The most common form of nudging is to present facts in such way that a certain outcome becomes more likely, and appealing to feelings of responsibility and duty is another common form. Some nudging in screening is inevitable and could be justified because of its beneficence if there is an obvious benefit-harm ratio. Nudging could be regarded as paternalistic since it implies that some kind of manipulation is needed to make rational decisions. Ethically sound nudging does not push people in a certain direction, but preserves their self-determination and helps them to choose according to their own values [48].
There is no consensus about what constitutes neutral information for informed consent before screening. Those who are skeptical about screening, which they regard as harmful and leading to expanding disease definitions and overmedicalization, obviously want to include
information about factors like the low probability for each individual to benefit, false positives and the uncertainty of planned interventions. Screening advocates are obviously more interested in the arguments for screening listed above. Other areas of disagreement are inevitably thresholds for what is abnormal, cutoff points for action and, most of all, who should decide these matters.
The evaluation of benefit and harm differs between both clinicians and patients and it seems unlikely that any given information should be able to satisfy everyone. Information which
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would be regarded as objective by vascular surgeons would most likely be regarded as totally biased by Johansson and vice versa.
An Australian study using a decision aid which included information on overdiagnosis when screening for breast cancer found that although there was a small decrease of enthusiasm for mammography most women remained positive [49]. Johansson found this study ethically unacceptable since the control group got less information about the harms of the intervention
[45].
A UK population survey of attitudes toward cancer screening found a wide variety of
conclusions among 1,000 respondents who were given the same information. Some were not willing to accept any overdetection, even if the potential benefits were presented as large. Others would tolerate overdetection in every screened individual [50].
The amount of information given will have to be individualized and the patients should be able to control how much information they need in order to make a decision. Some patients require information about technical details which would be overwhelming to others. A patient who decides that limited information is enough to make a decision has not been deceived
[51].
According to Brownsword, AAA screening which emphasizes individual rights by respecting autonomy and provides reliable informed consent results in benefit overweighing harm. The autonomy and rights of those unfortunate men who “end up in early graves” after making their own informed choices have not been violated and “there is no need for an ethical apology” [38].
Integrity
Autonomy is the capacity for rational decision-making in a competent person and thus related to what we have or possess. Loss of rational capacity means a loss of autonomy. Integrity, which comes from Latin ‘integer’, which means wholeness or complete unity, is related to value and dignity and what we are. It cannot be transferred to someone else and it cannot be lost, even if we lose our autonomy, since it is shared by all humans as a part of our being [52]. For Beauchamp and Childress integrity is a virtue related to a person’s moral character [53], but the basis for this discussion is physical and mental integrity as defined by the Swedish National Council on Medical Ethics. Physical integrity means that no one has the right to examine somebody else without the other person’s consent. Mental integrity means that an individual’s opinions and values may not be violated [54].
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Physical illness threatens the unity of the body, that is the integrity, which often means that the affected part feels alien and even like an enemy. Psychological illness means that anxiety, obsessions and other disorders control and distort existence. Serious illness alters self-image and confronts us with disability, pain and the possibility of nonexistence. In this situation the physician must be sensitive to the patient’s dependent and vulnerable state, and show respect for the patient’s values and choices [52].
An invitation to AAA screening may be an unwelcome reminder of our mortality and an AAA diagnosis is an obvious threat to our very existence. A previously healthy individual starts regarding himself as a patient, which is a kind of medicalization. Refusal to participate in screening could be a logical decision based on a rational opinion but it could also be a denial of the inevitable negative parts of life. No matter what the reason is or even if there appears to be no reason the patient’s decision should be respected because it is impossible to know the self-image, history and background of such a decision. However, there should always be an aim to work for the ideal, or perhaps utopian, situation in which the patient and the doctor work together towards a decision which both protects the health and the integrity of the patient [52].
There are few studies which touch on the subject of psychological effects in connection with AAA screening. There are some studies and systematic reviews which conclude that there is not enough data to provide accurate assessment of the frequency and extent of psychological effects when a person is screened with a positive result, and the general effects of screening in individuals with negative results have not been determined [55]. A UK study from 2018 showed a temporary reduction in mental quality of life after being diagnosed with AAA, which returned to normal levels after 12 months. The results are similar to screening programs for breast, prostate, and colon cancer [56].
Justice
Ethical justice in health care can be defined as a fair distribution of scarce resources. It could also mean to treat others equitably, respect for people’s rights and morally acceptable laws. Johansson argues that scarce resources should be spent on those who are already ill and have the greatest need instead of trying to maximize informed consent among healthy individuals
[45]. However, recently several researchers have published meta-analyses and long term evaluations which have shown that AAA screening is cost-effective.
18
The 25-year follow-up by Oliver-Williams noted that although decreased smoking rates probably explain much of the reduction of AAA and other degenerative vascular conditions, such as stroke and myocardial infarction some other factors remain unexplained. It was concluded that AAA screening remains cost-effective down to a prevalence of 0.35%, which is one-third of the current rate [30].
A 2018 Australian meta-analysis of five studies with 175,085 participants and a mean of 10.6 years of follow-up found a decrease of AAA-related mortality and all-cause mortality. 209 to 769 men needed to be screened in order prevent one AAA-related death. A wider
implementation of population-based AAA screening was recommended and found to provide a ‘significant and cost-effective’ reduction of AAA and all-cause mortality [57].
A 2018 article in Angiology discussed whether screening for AAA is a luxury or a necessity. Johansson’s claims that AAA screening causes more harm than good despite the robust data on the benefits was rejected except for specific categories of patients, e.g. those who have several comorbidities and/or are at high-risk for surgery. The cost of per life-year saved was USD 1173, which is less than screening for breast, cervical and colorectal cancer [58].
Limitations
First, this study consists of two large components, the scientific background and the ethical discussion, and a limitation was the number of references and words allowed. Without this limitation, a more detailed background and discussion, e.g. including patients’ experiences of AAA screening, could have been carried out. A second limitation was the online search for scientific articles, which was mainly made using PubMed and Google Scholar. Also, the debate comments were only obtained from one source. A wider, more general search might have lead to the discovery of more diverse opinions, thus this study has a selection bias. A third limitation was the inevitable interference of the author’s pre-understandings and preconceptions. A study of this nature cannot lead to a totally objective conclusion or an absolute truth.
Conclusion
AAA screening provides an interesting example of how modern medicine, although regarded as a part of natural science, is far from the exactness and objectivity of mathematics, physics and chemistry. Many medical outcomes are uncertain which partly explains why the
suggested treatment options vary between physicians. Even with this background it seems surprising that results from the same randomized controlled trials may be interpreted in very
19 different ways by different groups and individual actors. As we have seen, this may be related to different statistical methods and cutoffs, but also an all too human tendency to search for facts which support preconceptions rather than truth.
AAA screening should rely not only on relevant medical facts but also having a reliable ethical compass. A screening process which provides high quality informed consent does not violate the rights of the unfortunate man who may die on the operating table because his autonomy and integrity are respected.
Finding the optimal balance between benefits and harms in a constantly changing world is a challenge which requires an open mind. Resisting overdiagnosis is an obvious concern, but there are always two sides of the coin. Both action and inaction produce winners and losers and to choose not to act also has moral consequences. The major burden of proof has
rightfully been placed on the advocates of AAA screening but it should not be forgotten that a discontinuation of AAA screening will result in a number of otherwise preventable deaths from ruptured aneurysms. The autonomy and integrity of those men who die as a result of being denied AAA screening have also been violated. They are not killed on the operating table but by our decision not to offer them screening.
The different attitudes about AAA screening highlight very divergent views about the suitable limits of medical care. The controversy about AAA screening seems to reflect a pattern of irreconcilable convictions not only about medicine but also about other aspects of life, which are beyond the scope of this study. One extreme leads to overmedicalization and the other to nihilism and neither position benefits patients. Eagerness to convince may overshadow willingness to find truth, but it is of the utmost importance to remember that the ethically acceptable position of every physician should not be to boost one’s ego but to work for the best possible health for his or her patients.
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24
Appendix
Evaluation criteria for screening according to The National Board of Health and Welfare in Sweden
1. The condition should be an important health problem
The condition that the screening program intends to detect should lead to severe consequences, e.g. preterm death, severe lesion, or disability. If early interventions could lead to saved resources or less suffering, screening can also be made for
conditions with less severe consequences as long as they are important for the society or the individual. The consequences of the condition should be an important health problem. It could be important in different ways for the individual and society.
2. The natural course of the condition should be known
The natural course, and most of all its development from latent to symptomatic disease, should be described on a group level. In the case of non-progressive
conditions, the consequences should be known. The condition should lead to severe consequences for the majority of the group if no intervention is initiated.
3. The condition should have an asymptomatic detectable phase
The condition should have a detectable latent or asymptomatic phase. Diseases with a fast progression can have a short latent or asymptomatic phase. The benefit is reduced if the screening is performed too early or too late. A longer asymptomatic phase increases the risk of screening leading to overdiagnosis and overtreatment of conditions that would have never led to preterm death, severe lesion, or disability.
4. There should be an appropriate test method
The test method should have a scientifically proved ability to find or exclude the condition with high precision. The test method should have been evaluated in the intended population. If there is a need for complementary tests after an initial positive screening result, to ensure the diagnosis or the need for interventions, these test methods should also be well established and evaluated. The test result’s cut off values for subsequent interventions should be well defined.
25
5. Interventions should provide better effect at an early stage than if detected clinically
In order for the screening to be valid the interventions must be effective, and the effect should be better in an early phase than if there are symptoms. The negative effects of the intervention should be considered. Interventions performed at the time of
screening are sometimes associated with less harm than interventions performed after clinical discovery.
6. The screening program should decrease mortality, morbidity, or disability associated with the condition
The effect of the screening program should have been evaluated in well performed randomized population based studies. Some screening programs cannot be evaluated through randomized studies out of practical or ethical reasons. For these an alternative could be documentation from multiple, independent, and scientific studies about the processes with and without screening.
7. The test method and the following investigation should be accepted by the intended population
The majority of the intended population should accept the time consumption of the test, and regard the physical and psychological effect of further investigation as reasonable.
8. Interventions for the condition should be made clear and be accepted by the intended population
There should be generally accepted guidelines for the intervention of the condition when it is detected through screening. The interventions must be accepted by the majority of the individuals with the condition, with or without symptoms. There are few benefits with early diagnosis if a large proportion of the individuals are not subject to the recommended interventions until they have symptoms.
9. The health benefits should outweigh the negative effects of the screening program
Screening has both positive and negative effects. To be able to evaluate the balance between benefit and harm for each screening program, these effects should be highlighted. From an ethical perspective this balance is central – to be able to
26 recommend a screening program the positive effects must outweigh the negatives.
10. The screening program should be ethically acceptable
The screening program should be ethically acceptable regardless of whether it is found to have health benefits that outweigh the negative effects, is accepted by the
population, or has a reasonable cost-effectiveness. This requires a more extended ethical analysis that can include evaluations of
- The handling of possible negative effects, autonomy, and integrity - Long-term impact on human dignity and equality
- Groups with special values and interests
- Fair distribution of healthcare resources compared to other alternatives - Possible redistribution of responsibility and role between health care
professionals and the individual and how this can be managed - Impact on continued research in the field
- Legislations and other guidelines which provide direction for ethical evaluations concerning the above mentioned points
11. The cost-effectiveness should have been assessed and evaluated as fair in relation to the need
An analysis of the cost-effectiveness of the screening program should include information about costs and health effects. Cost-effectiveness is measured through a comparison between screening and one other relevant alternative (e.g. no screening, other intervals, or other age groups). The effects of screening can be life-years or quality adjusted life-years (QALY) gained. The cost can be evaluated by an estimated cost per QALY gained. The evaluation of the ethical platform should be done in relation to the severity according to the needs principle. When central data are
missing, an evaluation of cost-effectiveness is usually based on results from scientific research and suppositions.
12. Information about participation in the screening program should have been assessed
Validated, brief, and written information about the screening program should be given to all who are offered to participate. The information should clarify
27 - How the screening program affects consequences of the condition
- How the screening test is performed
- The proportion that gets a positive result of the screening test - The risk of a false positive or false negative result
- Consequences of a positive result of the screening test - Likely interventions and their consequences
- That participation in a screening program is optional - Who the sender is
- How one can acquire further, more detailed information
The information should be phrased in a simple and comprehensible way and should be available in the languages common among the intended population. Other aspects of the information that can contribute to an equal attendance should be considered. The information should be tested on the intended population before it is used. The need for information about the screening program to others than the intended population, e.g. health care providers, decision makers, and the general public should also be
evaluated.
13. Relevant organizational aspects should be clarified
There should be an organizational plan how to obtain national equivalence. The plan should comprise all elements of the screening program that are central to obtaining the intended effect. The plan should also specify the parts of the screening program’s organization which could be adjusted to local conditions. For example, the plan should include information about required equipment and expertise, and how the invitation to the program should be managed.
14. The screening program’s need for resources and practicability should have been assessed
The need for both economical and personnel resources should be assessed and described. The possible need for special resources or expertise that can arise due to more cases being early detected should be assessed. The resource needed to
investigate and give a clean bill of health to persons who have been afflicted by false positive screening results should be evaluated. There should also be a general analysis of possible obstacles for nationwide screening introduction. The present situation of ongoing local or regional screening programs and opportunistic screening should be
28 assessed.
15. There should be a plan for evaluation of the screening program’s effects
It should be possible to evaluate if the expected health benefits of the screening
program have been accomplished. A plan for evaluation and follow-up of the different parts (test, diagnosis, and treatment), as well as the entire screening program should exist before the start of the program. Both positive and negative effects should be followed. If a screening program’s conditions are changed, a new evaluation of the program should be done.
29
Populärvetenskaplig sammanfattning
Bråck på stora kroppspulsådern i magen innebär en lokal
utvidgning på kroppspulsådern på minst tre centimeter på grund av en försvagning i kärlväggen. Detta är fyra till sex gånger vanligare hos män. Bråcket ger oftast inga symtom, men om det brister kan det vara livshotande. I Sverige finns ett
screeningprogram för 65-åriga män där man använder ultraljud för att kunna upptäcka bråck. Denna uppsats granskar
screeningen ur ett etiskt perspektiv.
Det finns för- och nackdelar med screeningprogrammet vilket har lett till en intensiv debatt. Fördelarna är att ultraljudsundersökningen går fort, är smärtfri och är mycket tillförlitlig. Screening kan minska dödligheten med 40-50%. Socialstyrelsen och många forskare rekommenderar screeningprogrammet. En av nackdelarna är att man kan känna oro och ångest om man får en oväntad bråckdiagnos. En diagnos betyder inte säkert att bråcket kommer brista. En förebyggande operation kan ge komplikationer och i 1% av fallen leda till döden. Vissa forskare menar därför att screeningen borde ifrågasättas.
Man kan fråga sig om det är etiskt försvarbart att rädda livet på vissa medan andra, även om de inte är så många, dör. Därför är det mycket viktigt att ge korrekt och balanserad
information så att var och en själv kan bestämma om han vill delta i screeningen. Avgörande för beslut kommer i många fall inte bara vara fakta utan även livssyn och känslor. Detta måste respekteras av vårdgivaren även i de fall när man anser att beslutet inte är rationellt, eftersom det personliga självbestämmandet är viktigast av allt.
30
Cover letter
Örebro, Sweden. December 11, 2018 Dear John McMillan,
I am delighted to submit a manuscript study titled “Abdominal Aortic Aneurysm Screening – an Ethical Discussion” for consideration for publication in the Journal of Medical Ethics. The subject is more controversial than ever due to a 2018 dissertation that created a lively debate by questioning the value of the screening program in Sweden.
My manuscript offers not only a solid scientific background and a summary of the main arguments but also a thorough ethical analysis based on common bioethical principals. There are four main messages. First, this study shows that intellectual passion has created an unusually polarized discussion. Second, it seems that benefit outweighs harm. Third, since AAA screening is the first screening program which could lead to the death of a previously asymptomatic individual, well founded informed consent is extremely important. Fourth, both decisions to act and not to act have moral consequences.
I believe that this manuscript is suitable for publication by the Journal of Medical Ethics and that it will generate reflections for health care professionals and policy makers.
This manuscript has not previously been published and is not under consideration for publication elsewhere. All authors have approved the final manuscript and we have no conflict of interest to disclose.
Thank you for your consideration!
Sincerely,
Ami Holmström, Bachelor of Medicine School of Medical Sciences
Örebro University Örebro
31
Etisk reflektion
Detta är en kvalitativ litteraturstudie med hermeneutisk analys av text, argument och etik. Innehållet har i huvudsak hämtats från vetenskapliga artiklar. Varken studierna eller de texter som har analyserats innehåller några personuppgifter. Även om huvudinnehållet i arbetet är en etisk reflektion kring screening av bukaortaaneurysm har utarbetandet i sig inte varit förenat med något behov av etiska överväganden.
Framsteg kräver nytänkande och ifrågasättande av etablerade sanningar. Det är därför inte automatiskt förenat med etiska problem att forskare drar olika slutsatser av sina (eller andras) arbeten och ifrågasätter screeningmetoder. I de artiklar jag har läst verkar det vanligaste sättet vara att utifrån egna (eller andras) arbeten presentera medicinska fakta som, utan att man direkt uttalar det, visar att den granskade screeningen inte tillräckligt uppfyller de etiska principerna om nytta, undvikande av skada och rättvisa. Synpunkter på den för etiker
viktigaste principen om autonomi och informerat samtycke tycks mindre vanliga. Tendens till systematiskt urval av fakta som gynnar den egna åsikten verkar tyvärr inte vara helt ovanliga, vilket minskar möjligheterna till ett utvecklande meningsutbyte. Ett förslag till två frågor som forskare i kontroversiella frågor skulle kunna ställa sig för att förhoppningsvis höja den etiska nivån är: Är jag beredd att ändra min åsikt om nya fakta presenteras? Vilket är viktigast för mig, att vinna debatten eller att komma fram till sanningen?
