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BMJ 2016;353:i1156 doi: 10.1136/bmj.i1156 (Published 20 April 2016)
Page 1 of 5
Analysis
ANALYSIS
Harms and benefits of screening young people to
prevent sudden cardiac death
Sudden cardiac death of young athletes needs to be avoided but does screening really help? Hans
Van Brabandt and colleagues look at the evidence
Hans Van Brabandt researcher, Anja Desomer researcher, Sophie Gerkens economist, Mattias
Neyt health economist
Belgian Health Care Knowledge Centre (KCE), Brussels, Belgium
Sudden cardiac death of a young person on a sports field is a
devastating event. Often these deaths are due to an unrecognised
underlying heart condition, and screening has been proposed as
a method to prevent them. However, disagreement remains
about its benefits and harms.
In Italy, screening before participation in competitive sport has
been mandatory since the 1970s. But the Netherlands abandoned
its mandatory programme in 1984 because of the poor diagnostic
performance of screening tests.1 In the UK, Cardiac Risk in the
Young (CRY), a charity offering support and counselling to
affected families, runs screening events, but the National
Screening Committee decided last year not to recommend a
population screening programme.2 Here, we summarise the
findings of our literature review on the benefits and harms of
pre-participation screening for non-professional athletes aged
14-34 years.3 We defined athletes as anyone participating in an
organised team or individual sport that involves regular
competition against others, places a high premium on excellence
and achievement, and requires some form of systematic training.4
How common is sudden cardiac death in
young healthy athletes?
Sudden cardiac death may be caused by a panoply of rare genetic
and acquired heart disorders, present in 0.3% of the population.5
Most affected people are not aware of the disease and lead a
normal life, but some develop symptoms such as dizziness,
fainting, or breathlessness, which leads to a diagnosis. In around
1% of young athletes with unrecognised heart disease, sudden
death will be the first and only manifestation of the disease,6-8
and it is these people that screening aims to detect. Although it
is uncertain whether sudden cardiac death occurs more often
during exercise than at rest, research has predominantly focused
on athletes because exercise is considered a potential trigger.9
The exact frequency of sudden cardiac death in young athletes
is not known. Numbers obtained from heterogeneous populations
of general, college, and high school athletes indicate incidences
varying between 1 per 917 000 and 1 per 3000.10 A recent review
by the UK National Screening Committee suggested an annual
incidence of 1 per 100 000 people aged 12-35.11 Most of those
affected are men.12One UK study among athletes aged 12-35
years reported 64 deaths a year.13 In the UK National Audit of
Sudden Arrhythmic Death Syndrome, a yearly average of 36
cases are registered.14 The charity CRY claims “at least 600”
cases every year based on data from the Office for National
Statistics,15 but this includes all people aged 1-34 years, whether
or not they were athletes or had known heart disease.
Hypertrophic cardiomyopathy is the most common underlying
cause of sudden cardiac death in young people, but the absolute
risk of sudden death in people affected by the disease is low.16 17
In a prospective study in England, the annual incidence of
sudden death due to hypertrophic cardiomyopathy was around
0.5-1 per million inhabitants.6
How accurately can pre-participation
screening identify those at risk?
The most basic type of screening, as recommended by the
American Heart Association,18 comprises taking a personal and
family history in combination with a physical examination.
However, very few people at risk of sudden cardiac death can
be detected in this way.19 Of 115 young athletes who died
suddenly and who had had a standard pre-participation medical
evaluation, only four (3%) were suspected of having heart
disease, and the abnormality responsible for the death was
correctly identified in only one athlete (0.9%).20 To increase the
diagnostic yield of screening, some experts advocate including
a resting electrocardiogram (ECG).21 This may allow for better
prediction of certain arrhythmias, such as those associated with
Correspondence to: H Van Brabandt [email protected]
Data supplements on bmj.com (see http://www.bmj.com/content/353/bmj.i1156?tab=related#datasupp)
Table evaluating diagnostic performance of screening
For personal use only: See rights and reprints http://www.bmj.com/permissions
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BMJ 2016;353:i1156 doi: 10.1136/bmj.i1156 (Published 20 April 2016)
Page 2 of 5
ANALYSIS
the Wolff-Parkinson-White syndrome. However, athletes with
a congenital anomaly of the coronary arteries or with premature
coronary artery disease will not be identified by
electrocardiography. Overall, 25% of people affected by a
disease that may lead to sudden cardiac death would remain
undetected.3 16 In other words, the sensitivity of a screening
examination that includes electrocardiography is 0.75.
Correctly identifying athletes with a disease that may lead to
sudden cardiac death (true positives) is only one side of the
diagnostic medal. Screening may also lead to inappropriately
suspecting disease in healthy people (false positives). In a recent
US study of 1339 students, 916 (68%) reported at least one
positive response on a questionnaire with heart health questions.
After review by a physician, 421/1339 (31.4%) had at least one
relevant response that required additional cardiac evaluation.22
Electrocardiography will pick up only some of the possible
causes of signs and symptoms. For example, the resting ECG
may be normal in someone with a heart murmur or who
mentions that a syncope, palpitations, or chest discomfort has
ever occurred during or shortly after exercise. Yet, these clinical
findings may be a sign of a disease that can lead to sudden death
and can therefore not be ignored. Based on data from small case
series we estimated a specificity for screening including history,
physical examination, and electrocardiography, ranging from
0.70 to 0.95.3 If screening is performed by experienced
physicians, even with the application of the most stringent
“Seattle” criteria for electrocardiography, 5% of healthy people
will be suspected of having disease.23-25 In a more realistic
scenario, up to 30% of those screened may be referred for
additional testing after screening.19
The picture is further complicated by the clinical presentation.
Most people with a cardiac disease are asymptomatic or have
no specific clinical signs.26-28 Symptoms that may indicate serious
disease (such as dizziness, syncope, palpitations, and chest
discomfort) are aspecific and more often caused by other more
benign conditions. Furthermore, there is a large overlap of the
normal ECG and the ECG of someone with one of the index
cardiac diseases. This overlap is even more pronounced in highly
trained athletes, whose ECGs may show physiological alterations
that mimic disease.
What is the benefit of pre-participation
screening?
To reliably assess whether screening can reduce the number of
sudden cardiac deaths in young athletes requires a randomised
controlled trial.29 30 Unfortunately, no such study exists. The
only evidence that screening saves lives comes from a
population based study in Italy.5 31 A 90% reduction in the
incidence of sudden cardiac death in young athletes was
observed in the Veneto region over 26 years after mandatory
screening was introduced in 1979 (from 3.6/100 000 person
years to 0.4/100 000 in 2004; fig 1).31
Some experts have argued that this study does not provide
evidence that screening was the cause of the fall in incidence.32
Since there is no unscreened control population, the fall may,
for example, have been due to improved resuscitation or to an
increasing proportion of female athletes, who are known to be
much less prone to sudden cardiac death. Furthermore, the
remarkably high incidence at the start of the study (3.6/100 000
person years) was calculated on no more than 14 cases and
might represent mere random variation.33
The fact that the incidence of sudden cardiac death in countries
with no mandatory screening such as the US (Minnesota) and
For personal use only: See rights and reprints http://www.bmj.com/permissions
France is no different from the most recently reported data from
Italy (fig 1) further suggests that the observed fall in Italy was
not the result of screening.12 34
What are the harms of screening?
Based on the diagnostic performance of screening discussed
above, we can assume that out of one million young people
screened, 50 000 to 300 000 will be identified as being suspected
of having a disease that may lead to sudden cardiac death (see
supplementary data on thebmj.com).
They will need additional cardiovascular testing, which may
cause anxiety and psychological harm, unnecessary and
unwarranted restrictions and disqualifications from sports, and
impediments to insurability or employment opportunities.32 35
According to bayesian logic,36 a hyperoptimistic scenario
assuming exceptional performance of downstream investigations
(sensitivity of 1.0 and a specificity of 0.99 as shown in the
supplementary table on thebmj.com) will end up with up to
5200 out of a million people screened being labelled for the rest
of their life with a potentially lethal diagnosis. In the Italian
pre-participation screening programme, 2% of people screened
(20 000/million) were ultimately disqualified from participation
in competitive sports.31
The most prevalent diseases detected at screening are the
Wolff-Parkinson-White syndrome (550-2180/million),37
congenital anomalies of the coronary arteries (1000/million),16 38
and hypertrophic cardiomyopathy (100-790/million).37 Most
people with these diseases will never experience any symptoms
and lead a normal life if the disease is left undetected.26-39 There
is no consensus on treatment in asymptomatic individuals. Some
argue that they do not need to be treated; others will proceed to
catheter ablation or the insertion of an implantable defibrillator.
However, the risk of death associated with these treatments is
similar to that of sudden cardiac death in asymptomatic
people.4-41
Call for open data
During our study of the literature on screening, we noticed that
the Italian researchers who published the Veneto study
passionately promote screening. Through repetition of their
study results in secondary papers, and their participation in
guideline development groups and consensus meetings, they
have had an important role in convincing sports physicians and
sports federations around the world to introduce pre-participation
screening.31-45 In their seminal paper they concluded that “The
incidence of SCD in young competitive athletes has substantially
declined in the Veneto region of Italy since the introduction of
a nationwide systematic screening.”31 However, over the years,
and with no new evidence, the wording has gradually changed
and increasingly suggests a causal relation. In a 2013 paper they
wrote: “Pre-participation screening based on a 12-lead ECG is
effective in identifying athletes with potentially lethal
cardiovascular disease and saves lives.”43 In addition, the Italian
investigators claim that guidelines from the European Society
of Cardiology (ESC) recommend pre-participation screening
with electrocardiography.44-48 However, the document they refer
to is in fact a “proposal for a common European protocol” of
which they are the principal authors.21 Furthermore, they suggest
that screening is cost effective by referring to economic models
that used their data as input variables.42
Some of the concerns about the Veneto study might be clarified
if the Italian investigators would provide access to additional
unpublished data. Although pre-participation screening is
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BMJ 2016;353:i1156 doi: 10.1136/bmj.i1156 (Published 20 April 2016)
Page 3 of 5
ANALYSIS
mandatory throughout Italy, only data from one region (Veneto)
have been published. Furthermore, since screening in Italy
became mandatory in 1971, data collected before 1979 should
be available showing whether the unexpectedly high initial
incidence was simple random variation. Italian data obtained
after 2004 have also not been published. These missing data
would allow better understanding of the consistency of the initial
findings.
Researchers from the UK tried to obtain additional data from
the Italian screening programme through a formal request from
Jeremy Hunt, the secretary of state for health for England, to
the minister of health in Italy, to no avail.11 Our requests to the
Italian researchers in June 2014 and January 2015 also remained
unanswered.
Conclusion
The effectiveness of pre-participation screening in reducing the
number of sudden cardiac deaths among young athletes has not
been substantiated by solid evidence. Its potential to reduce
deaths is likely to be low because of the poor detection rate and
the uncertain effectiveness of the management of the diseases
thus identified in asymptomatic people. Pre-participation
screening induces harm because of the high number of false
positive test results leading to temporary or lifelong
disqualification from competitive sports, psychological and
financial harm, and medical follow-up and treatment with
unknown benefit.3-36 In addition, it leads to young people
avoiding exercise known to be beneficial to their overall health.
As long as those at high risk of sudden death cannot reliably be
identified and appropriately managed, young athletes should
not be submitted to pre-participation screening.
Contributors and sources: This article is the summary of a Health
Technology Assessment on cardiovascular pre-participation screening
in young athletes, produced by the Belgian Health Care Knowledge
Centre (KCE) and commissioned by the Belgian authorities. The full
text can be accessed online (https://kce.fgov.be/publication/report/
cardiovascular-pre-participation-screening-in-young-athletes). HVB is
a cardiologist with 30 years’ clinical experience. He is a former director
of the Belgian Centre for Evidence-Based Medicine and Cochrane
Belgium. AD has a master’s degree in rehabilitation sciences and has
been involved as researcher in health services research projects and
in the development of clinical practice guidelines. SG is involved in
health technology assessments and health services research projects
and is a member of the Health Systems and Policy Monitor network.
MN contributed to the development of the Belgian guidelines for
economic evaluations and budget impact analyses and the European
guidelines for health related quality of life and utility measures. He is
cofounder of ME-TA, which offers expertise to foreign non-profit
organisations and provides training and education in health technology
assessment and economic evaluations.
Competing interests: We have read and understood BMJ policy on
declaration of interests and have no relevant interests to declare.
Provenance and peer review: Not commissioned; externally peer
reviewed.
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BMJ 2016;353:i1156 doi: 10.1136/bmj.i1156 (Published 20 April 2016)
Page 4 of 5
ANALYSIS
Key messages
An estimated 0.001% of young athletes die suddenly every year, and pre-participation screening aims to detect heart disease that puts
them at risk
Up to 30% of those screened may be referred for additional testing, leading to unnecessary anxiety, overdiagnosis, overtreatment, and
exclusion from sport
Screening would not detect around 25% of those at risk of sudden death and no randomised trial has examined its effectiveness
On balance cardiovascular screening in young athletes is likely to be more harmful than beneficial
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Accepted: 15 02 2016
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BMJ 2016;353:i1156 doi: 10.1136/bmj.i1156 (Published 20 April 2016)
Page 5 of 5
ANALYSIS
Figure
Annual number of sudden cardiac deaths per 100 000 athletes in Veneto (Italy),31 France,12 Minnesota (US),34 and Israel.33
Screening became mandatory by law in Italy in 1971 and in Israel in 1997 (arrow). Graph adapted from Steinvil et al33
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