Development of a parental information pamphlet for

rheumatic heart disease echocardiography screening

Brief Study Summary and Discussion of Focus Group Recommendations

Citation: Jason Gurney, Virginia Signal and Diana Sarfati (2016). Development of a parental
information pamphlet for rheumatic heart disease echocardiography screening: Brief study summary
and discussion of focus group findings. Wellington: University of Otago.

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This document is intended to compliment the focus group report produced by CBG Health Research
Ltd., which provides the key findings from the focus group workshop. This report can be viewed at
http://www.otago.ac.nz/wellington/departments/publichealth/research/cancercontrol/projects.

The current document provides a brief background and summary of the study, and then details
changes made (or reasons why changes were not made) following the recommendations of the focus
group.

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Acknowledgements
We would like to thank Prof Jonathan Carapetis, Prof Michael Baker, Dr Nigel Wilson and Dr Caroline
Shaw for providing expert feedback on the content of the pamphlet. We would also like to thank CBG
Health Research Ltd. for convening the focus group, and the participants for giving their valuable time
and perspective.

Funding
This study was funded by the Health Research Council (reference # 13/965), the Ministry of Health, Te
Puni Kōkiri, Cure Kids and the New Zealand Heart Foundation.

Ethical Approval and Consultation

This study received ethical approval from the University of Otago Human Ethics Committee (reference
# D16/168). Consultation was also undertaken with the Ngai Tahu Research Consultation Committee.

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Introduction
Each year, more than 150 New Zealanders die of rheumatic heart disease (RHD). 1 New Zealanders of
Māori and Pacific ethnicity are up to ten times more likely to die of RHD than non-Māori/Pacific New
Zealanders.1 2 The prevalence of RHD peaks in the 20-59 year age group,3 while mortality from this
disease is most likely to occur in middle age (mortality rate for 50-59 age group: Māori 28 per
100,000; Pacific 18 per 100,000, non-Māori/Pacific 1 per 100,000).1

RHD is the cardiac consequence of acute rheumatic fever (ARF). At the root of ARF is an immune
response to streptococcal infection (typically Group-A streptococcus, or GAS), the results of which
may include joint pain, rashes, abnormal movements (chorea) and endocarditis. It is the last of these
consequences which may permanently damage heart valves, and it is this manifestation which is the
primary characteristic of RHD.3 4 This valvular damage can lead to debilitating chronic heart disease,
heart failure and consequent increased risk of mortality.3

Screening to reduce the burden of RHD

Beyond the primary prevention of ARF, a reduction in the burden of RHD may be possible by
identifying the condition while the patient is still asymptomatic and then intervening with secondary
antibiotic prophylaxis, or with cardiac surgery if severe disease is observed. It is estimated that 40%
of patients who present with symptomatic RHD do not have a known history of ARF, 5 and it is these
patients who should be the target of population screening. Screening aims to identify these patients
before they become symptomatic, and therefore preventing ARF reoccurrence via secondary
prophylaxis. The goal is to reduce the likelihood that these patients progress to a severe,
symptomatic stage of RHD and thus be less likely to require costly and risk-filled intervention.6 There
are obvious benefits of early-detection among those who would have progressed to severe,
symptomatic RHD – including the possible prevention of substantive morbidity and early mortality.

It has been suggested that “all screening programmes do harm; some do good as well”.7 There is
ongoing debate about whether it is socially and ethically appropriate to screen for rheumatic heart
disease (RHD) in a symptomless population. There are several reasons why this controversy exists,
relating primarily to uncertainty around the natural history of RHD, issues around who should receive
secondary prophylaxis, and diagnostic definitions of sub-clinical or ‘borderline’ RHD.8 Over-diagnosis
of questionable abnormalities (or ‘false-positive’) is of particular concern.

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Informing choice through increased health literacy

Ensuring that screening participants and their families understand the risks and benefits of rheumatic
heart disease (RHD) screening to better make an informed choice regarding participation is crucial in
ensuring the safety of the screened population. The aim of the current study was to develop an
educational tool to inform the screened population and their families about the likely balance of
benefits, potential harms and uncertainty around RHD screening.

Summary of Methods
Information pamphlet

We designed a one-page learning material (i.e. pamphlet) for circulation among present and future
screened populations and their families. This learning material was intended to have a mixture of
clinical- and screening-related content, and was constructed with experts from both these fields as
well as other relevant parties.

The learning material aimed to cover the following areas: a) description of the potential benefits of
screening, including improved health outcomes via early diagnosis of RHD; b) description of the
potential harms of screening, including over-diagnosis; and c) description of the uncertainty around
RHD screening, including the unknown natural history of symptomless RHD.

The pamphlet was not designed to address operational issues relating to the screening test itself. It is
envisaged that such information would be provided separately to prospective screening participants
and their family/ whanau.

We sought and gained feedback from clinicians, public health workers and researchers with expertise
in rheumatic fever, rheumatic heart disease and/or screening (JC, MB, NW, CS) regarding the
factuality and relevance of the material content. We then amended content based on this feedback.

Once the content of the information pamphlet was finalised, we designed and illustrated the learning
materials in Microsoft Powerpoint. We used exemplars from highly-effective materials published in
similar contexts, particularly material from the New Zealand Government’s Social Policy Evaluation
and Research Unit (‘Superu’).9

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Ethical approval and iwi consultation
We sought and gained approval for the focus group component of the study from the University of
Otago’s Human Ethics Committee (reference # D16/168). We also undertook consultation with the
Ngai Tahu Research Consultation Committee, who provided a letter of consultation.

Focus group

The usefulness of the pamphlet was qualitatively assessed using a focus group of parents. CBG Health
Research Ltd. (‘CBG’), an independent provider of public sector surveying with skills in convening
focus groups, assisted with identifying and recruiting focus group members from a sample of the
target population. We recruited 8 participants; 4 Māori, 3 Pacific and 1 NZ European, to (loosely)
reflect the incidence of Acute Rheumatic Fever (ARF) in New Zealand.10 11

Prior to the focus group, the study investigators collaborated with CBG to develop a pro-forma (or
discussion guide) to guide the focus group workshop, including a) questions pertaining to what
participants understand about ARF and RHD screening pre- and post-focus group, and b)
questions/discussion points relevant to the learning material itself. The final discussion guide is
included in the appendices of this brief report (Appendix 1).

CBG convened the focus group for a 2 hour workshop, and focus group participants were offered a
koha to cover costs of travel, time and/or childcare. Following the focus group, CBG prepared a full
report, highlighting the key findings and recommendations of the focus group.12

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Key Findings from the Focus Group Workshop
Findings from the focus group workshop are presented in detail in the CBG report.12 The key
recommendations made by the focus group were:

 The group liked the question style approach and section layout.

 The group advised that less-literate people would struggle to read the material, and
recommended revising the material to

a) increase the use of plain and simple language,

b) remove acronyms and technical terms,

c) replace with layman words, and

d) increase the use of personal language.

 The group observed that the learning material left doubt regarding whether the
parent/guardian should agree to have their child screened, and recommended removal of this
doubt and assurance of the need for this screening test.

When participants were told about the screening test before the focus group began (using the
description outlined at the beginning of the pro-forma/discussion guide), all eight were happy to have
their child undergo the test – because it was non-invasive, painless and free of cost. At the conclusion
of the focus group – after going through the pamphlet – half (4 out of 8) said they would still be
happy for their child to undergo the test.

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Response to Focus Group Recommendations
Based on the report produced by CBG,12 we have identified a series of recommendations made by the
focus group. These recommendations were taken into consideration by the study investigators, and
the information pamphlet was substantially revised as a result. A side-by-side comparison of the pre-
focus group and post-focus group pamphlets can be viewed in Figure 1.

Each of the recommendations is detailed below, followed by our response and details of relevant
changes made to the material.

Recommendation 1: The focus group made several recommendations with respect to making to the
material easier to understand, including increasing the use of plain and simple language, removing
technical terms and replacing these with ‘lay’ words, and increasing the use of ‘personal’ language.

 Response: We have made extensive changes to the wording of the pamphlet, including using
more plain language, removal of technical terms (and replacement with lay-terms where
possible) and attempted to increase the use of personal language. All of these changes have
both reduced the word-count and made the material easier to read and understand.

 For example, we have taken the recommendation of the focus group to simplify the
explanation of the text beneath the headings ‘What is RHD?’ and ‘Why screen for RHD?’, and
changed these to a more pointed bullet-point list involving more lay-language.

 However, there is also risk associated with simplifying a complex issue – and we have
attempted to strike a balance between accurate reporting of best-evidence and simplicity of
the language used in the material.

Recommendation 2: One of the main points raised by the focus group was as follows: “Overall
participants suggested the material requires mitigation of the doubts raised and increased assurance
about the necessity for, and outcomes from, the screening.”

The focus group makes recommendations “to lower doubt and increase certainty about the value of
the screen.”

 Response: Before the focus group occurred, the facilitators (CBG Health Research Ltd) were
informed that the objective of the pamphlet was not to convince people to undertake RHD

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 screening (i.e. ‘sell’ the screening), but rather to inform them about the benefits, harms and
uncertainties around screening. They were also told that the pamphlet was supposed to be a
balanced (lay) representation of the current state of evidence around RHD screening.

 Despite this, based on the key recommendation above (and many other recommendations
made in the report), it appears that the focus group were of the opinion that we should make
the pamphlet more unequivocal, by removing doubt and providing assurance regarding
whether someone should undertake screening. For example, one focus group member
commented: “It has to use more positives, install confidence, especially for something new.”
This reflects the commonly observed, and very powerful, intuitive appeal of screening that
assumes screening must be good.

 The objective of the learning material was to present the best evidence available regarding
the possible benefits, harms and uncertainties associated with RHD screening, so that
parents/guardians can make a relatively informed choice regarding whether their child should
take part. There is insufficient evidence to assure parents/guardians that screening is a good
idea (or not) – and this is reflected in the level of uncertainty presented in the learning
material.

Recommendation 3: “Participants did not identify anything culturally inappropriate. However, they
judged some Pacific and/or less literate people would struggle with the number of words and sentence
structure.”

 Response: As mentioned above, we have made extensive changes to the wording of the
pamphlet, which has both reduced the word-count and made the material easier to
understand.

Recommendation 4: A number of participants made comments regarding their confusion about what
they were supposed to take away from the pamphlet. Was screening a good idea, or was it a bad
idea? They were unsure.

“The group shared that overall the tone of the material seems to be one of doubt about the value of
the screen.“

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  Response: These comments reflect the high degree of uncertainty around RHD screening –
since those researching this topic are uncertain about the efficacy of this screening, it isn’t
surprising that a lay audience – when presented with the best knowledge that we have on the
subject – would also feel uncertain after reading this material.

 There are uncertainties about both the benefits and harms of RHD screening, so the fact that
participants identified this uncertainty after encountering the material may indicate a better
understanding of the issues than they had before encountering the material.

Recommendation 5: “This scan is likely to misdiagnose (the absence of misdiagnosis rate informed
ideas of ‘likely’ because if it is low, it would be stated).”

 Response: In the absence of a RCT of RHD screening, it is very difficult to estimate the extent
to which over-diagnosis might be occurring in the context of a population-based screening
programme. Thus, it is not possible to adequately address this recommendation.

 We have added an additional point to the pamphlet which states: “We need to learn more
about the benefits and harms of screening. For example, we are still uncertain how many
children might be wrongly labelled as having a problem when, in fact, there is not one.”

Recommendation 6: The focus group made recommendations to add “pictures that provide real life
connections to the messages, ideas included: RHD boy from the HPA advertisements, ECHO machine,
children being scanned or treated.

 Response: We have chosen not to include these pictures, but rather stay with neutral
symbols. Such pictures will be provided by the purveyors of the screening test, and as such
do not need to be part of the current material.

Recommendation 7: The focus group recommended changing the structure of the pamphlet to a fold-
out pamphlet, to “…help the sections be more discrete; allow the reader to concentrate on one part of
the information without the distraction of other sections; aid navigation and flow of the material;
(and) encourage retention of the material on the fridge or in a bag.”

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  Response: While there is certainly merit to having a fold-out version of this pamphlet, in the
first instance we would like to keep the current format – since it is conducive to online
viewing, which is where the pamphlet will first be made available. We may develop a fold-out
version of this pamphlet in the future.

Recommendation 8: The focus group recommended adding the words ‘a lot’ to the data presented
regarding the proportion of RHD patients who do not have a documented history of rheumatic fever.
“It would be good to say, a lot of people who have rheumatic heart disease did not know they had ever
had rheumatic fever. Some struggle with statistics.”

 Response: Rather than adding the words ‘a lot’ to the relevant section, we have replaced the
percentage (40%) with a phrase that is possibly more intuitive to understand: “Four out of ten
people who are admitted to hospital…”.

Recommendation 9: The focus group wanted more information about what is involved with the
screening test.

 Response: This is outside the objective of the current pamphlet. This material is supposed to
sit alongside other material that fully-explains what is involved in the test.

Recommendation 10: “The Pacific representatives associated the icon opposite with cutting cake which
they deemed distracting from the message.”

 Response: We have removed the pie chart from the pamphlet, and replaced with a simple
word-bubble that summarises the primary justification for screening (“Many people who have
RHD do not know that they have had rheumatic fever.”) While this statement is actually
ambiguous – since we do not actually know whether these patients knew that they had had
rheumatic fever – we have included it here because it explains the primary justification for
screening in an intuitive manner.

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Recommendation 11: The focus group members universally disliked the ‘icons’ that were used, and
made several comments to this effect.

 Response: In the ‘uncertainties’ section, we have replaced the majority of the icons with large
text. We have also changed other icons where relevant, but contrary to a recommendation
from the focus group we have retained the icon which shows a syringe. We believe this icon
quickly conveys the fact that treatment for definite RHD involves injections (rather than pills),
without needing to convey this information at length.

Recommendation 12: The focus group recommended removing the term “This sounds like a good idea
but…” from the pamphlet.

 Response: We have replaced this phrase with ‘However’.

Recommendation 13: The focus group recommended changing the title of the pamphlet, since it
involves two ‘negative’ words to one ‘positive’ word.

 Response: Since these are three key components that underpin this pamphlet, we disagree
with the focus group and have decided not to change the title.

Recommendation 14: With respect to the harms of screening, the focus group respondents
“questioned the necessity to state that the diagnosis will cause worry as it is a given.”

 Response: While worry surrounding an accurate diagnosis is indeed a given, the primary harm
associated with RHD screening is that not all children diagnosed will actually have the disease.
The worry experienced by parents who are (wrongly) told that their children have RHD is a
screening-related harm – and thus is important to include in the current material.

Finally, one of the comments from the focus group regarding the information provided about the
recent drop in rheumatic fever rates was quite concerning. One focus group member stated that
“Rheumatic fever is a problem of the past (45% drop in rates).” The pamphlet should not leave the
reader with the impression that rheumatic fever is a problem of the past – particularly since recent

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notification data from the Institute of Environmental Science and Research (ESR) shows a marginal
increase in first-episodes of RF between 2015 and 2016.13 Another reason for dropping this point is
that the issue of whether rates of RF are reducing or not at a population level is unlikely to
significantly affect a parent’s decision to allow their child to be screened for RHD.

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Figure 1: Side-by-side comparison of pre-focus group and post-focus group versions of the information pamphlet.

Front page – Pre-focus group: Post-focus group:

``
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(Figure 1, cont.)

Back page – Pre-focus group: Post-focus group:

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Conclusions
The information pamphlet that we have developed is unique, in that it is the first (to our knowledge)
attempt to present the current evidence regarding the benefits, harms and uncertainties of RHD
screening to a lay-audience. It has been amended based on critique from experts in the field of
rheumatic fever, rheumatic heart disease and population screening, and also a focus group of Māori,
Pacific and European parents.

We believe that we have presented the best-quality pamphlet possible, and that the information
presented adequately reflects the current state of evidence in this context. As can be noted from
both the focus group report and our responses to the focus group recommendations, there is
difficulty in balancing accuracy with simplicity in what is an inherently complex area. As more
evidence comes to hand, it may be possible to reduce the amount of uncertainty expressed in the
pamphlet. However, we believe that the current pamphlet will at least increase the probability that
the parents of children approached to undergo RHD screening will make a choice that is consistent
with their own values.

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References
1. Milne RJ, Lennon D, Stewart JM, Vander Hoorn S, Scuffham PA. Mortality and hospitalisation costs
of rheumatic fever and rheumatic heart disease in New Zealand. Journal of Paediatrics and
Child Health 2012;48(8):692-97.
2. Webb R, Wilson N. Rheumatic fever in New Zealand. Journal of Paediatrics and Child Health
2012;In Press.
3. Marijon E, Mirabel M, Celermajer DS, Jouven X. Rheumatic heart disease. The Lancet
2012;379(9819):953-64.
4. Roberts K, Colquhoun S, Steer A, Reményi B, Carapetis J. Screening for rheumatic heart disease:
Current approaches and controversies. Nature Reviews Cardiology 2013;10(1):49-58.
5. Carapetis JR, Currie BJ, Mathews JD. Cumulative incidence of rheumatic fever in an endemic
region: a guide to the susceptibility of the population? . Epidemiology and Infection
2000;124(239–244).
6. Reményi B, Wilson N, Steer A, Ferreira B, Kado J, Kumar K, Lawrenson J, Maguire G, Marijon E,
Mirabel M, Mocumbi AO, Mota C, Paar J, Saxena A, Scheel J, Stirling J, Viali S, Balekundri VI,
Wheaton G, Zühlke L, Carapetis J. World Heart Federation criteria for echocardiographic
diagnosis of rheumatic heart disease-an evidence-based guideline. Nature Reviews
Cardiology 2012;9(5):297-309.
7. Gray M, Patnick J, Blanks R. Maximising benefit and minimising harm of screening. BMJ
2008;336(7642):480-83.
8. Roberts K, Colquhoun S, Steer A, Reményi B, Carapetis J. Screening for rheumatic heart disease:
current approaches and controversies. Nature Reviews Cardiology 2013;10:49-58.
9. Social Policy Evaluation and Research Unit. Vulnerability in early life: How are New Zealand
children faring? In: Auckland NZGaTUo, ed., 2015.
10. Milne RJ, Lennon DR, Stewart JM, Vander Hoorn S, Scuffham PA. Incidence of acute rheumatic
fever in New Zealand children and youth. Journal of Paediatrics and Child Health
2012;48(8):685-91.
11. Jaine R, Baker M, Venugopal K. Epidemiology of acute rheumatic fever in New Zealand 1996-
2005. Journal of Paediatrics and Child Health 2008;44(10):564-71.
12. CBG Health Research Ltd. Development of a parental information pamphlet for rheumatic heart
disease echocardiography screening: Focus group testing – key findings. Auckand, New
Zealand, 2016:1-22.
13. Institute of Environmental Science and Research. Rheumatic Fever Report, July 2015 - June 2016:
Institute of Environmental Science and Research, 2016.

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Appendix 1: Focus group discussion guide
The document below was developed by the study investigators, in conjunction with CBG Health
Research Ltd. It was used to guide discussion during the focus group.

Thanks & We are here to provide feedback on this information pamphlet.

Introductions
Pre-pamphlet Questions to be asked BEFORE pamphlet is given:
understanding Your (healthy) child has brought a letter home from school, asking for your
of RHD consent to allow them to undergo a painless chest scan to look for
screening underlying heart problems. The test is free.

 Would you be happy for your child to undergo this test?

 Can you foresee any problems with having this test done?

Hand Out Pamphlets

& Invite Participants to read it.

Probes (if not covered)

First Look/Appeal Layout
Impressions Ease of reading Level of detail
Understandable Factual
Concise Culturally appropriate
Other comments Suggestions

What is the pamphlet talking about?

Probes (if not covered)

The benefits What will readers understand the benefits to be?

of screening? Does the information provide understanding of the possibility of improved
health outcomes via early diagnosis?
How (if at all) could this information attract people to screening?
Any improvements?

The harms of What will readers understand the harm to be?

screening Does the information provide understanding of:
 unnecessary diagnosis?
 likelihood of anxiety?
 possible changes in physical activity?
 the fact that the screening will not just test for RHD?
How (if at all) could this information deter people from screening?
Any improvements?

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Uncertainties What will readers understand about uncertainties?
around Does the information provide understanding of:
screening  uncertainty around diagnosis?
 the lack of treatment for borderline RHD?
 that RHD is a rare disease, and that rates of RF appear to be
reducing?
How will people receive this information?
How (if at all) could this information deter people from screening?
Any improvements?
Post-pamphlet Questions to be asked AFTER pamphlet is given:
understanding What are some of the benefits of RHD screening?
of RHD
screening What are some of the ‘costs’ of RHD screening?

Knowing what you know now, would you still be happy for your child to
undergo echo screening for RHD?

What extra information would you want to receive before allowing your
child to undergo RHD screening?

Did you find the learning material useful?

What did you find most useful?

Last Other comments/suggestions

Impressions

Thank & Close

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