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A worked example of 'bes fit' framework synthesis: A systematic review of views concerning the taking of some potential chemopreventive agents

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A variety of different approaches to the synthesis of qualitative data are advocated in the literature. The aim of this paper is to describe the application of a pragmatic method of qualitative evidence synthesis and the lessons learned from adopting this "best fit" framework synthesis approach. An evaluation of framework synthesis as an approach to the qualitative systematic review of evidence exploring the views of adults to the taking of potential agents within the context of the primary prevention of colorectal cancer. Twenty papers from North America, Australia, the UK and Europe met the criteria for inclusion. Fourteen themes were identified a priori from a related, existing conceptual model identified in the literature, which were then used to code the extracted data. Further analysis resulted in the generation of a more sophisticated model with additional themes. The synthesis required a combination of secondary framework and thematic analysis approaches and was conducted within a health technology assessment timeframe. The novel and pragmatic "best fit" approach to framework synthesis developed and described here was found to be fit for purpose. Future research should seek to test further this approach to qualitative data synthesis.
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RESEARC H ARTIC L E Open Access
A worked example of best fitframework
synthesis: A systematic review of views
concerning the taking of some potential
chemopreventive agents
Christopher Carroll
*
, Andrew Booth, Katy Cooper
Abstract
Background: A variety of different approaches to the synthesis of qualitative data are advocated in the literature.
The aim of this paper is to describe the application of a pragmatic method of qualitative evidence synthesis and
the lessons learned from adopting this best fitframework synthesis approach.
Methods: An evaluation of framework synthesis as an approach to the qualitative systematic review of evidence
exploring the views of adults to the taking of potential agents within the context of the primary prevention of
colorectal cancer.
Results: Twenty papers from North America, Australia, the UK and Europe met the criteria for inclusion. Fourteen
themes were identified a priori from a related, existing conceptual model identified in the literature, which were
then used to code the extracted data. Further analysis resulted in the generation of a more sophisticated model
with additional themes. The synthesis required a combination of secondary framework and thematic analysis
approaches and was conducted within a health technology assessment timeframe.
Conclusion: The novel and pragmatic best fitapproach to framework synthesis developed and described here
was found to be fit for purpose. Future research should seek to test further this approach to qualitative data
synthesis.
Background
While the potential limitations of qualitative data synth-
esis are frequently articulated, so is the utility of con-
ducting such analysis [1]. Framework synthesis is one of
several methodologies currently being developed for
synthesising qualitative data [2]. This type of synthesis is
based on framework analysis [3] and offers a highly
structured approach to organising and analysing data
(e.g. indexing using numerical codes, rearranging data
into charts etc.)[2]. It involves the preliminary identifi-
cation of apriorithemes against which to map data
from included studies. In contrast to such methods as
meta-ethnography [4], framework synthesis is primarily
a deductive approach. As such it carries certain
pragmatic advantages which might prove beneficial
within the constraints of a health technology assessment
where effectiveness review, economic evaluation and
qualitative evidence synthesis are conducted together
within tight time constraints. Thus a framework may
not simply be an instrument for analysis but may also
represent a scaffold against which findings from the dif-
ferent components of an assessment may be brought
together and organised. Limited numbers of published
examples of framework synthesisexist, among which
the most prominent have been produced by the same
team at the Institute of Education, University of London
[5-7]. The present synthesis therefore represents an
early worked example of this approach, the only one ori-
ginating from outside of the team who developed the
method, and offers an opportunity for further methodo-
logical advances. It is also the first to explore the
* Correspondence: c.carroll@shef.ac.uk
Health Economics and Decision Science (HEDS), School of Health and
Related Research (ScHARR), University of Sheffield, Sheffield, UK
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© 2011 Carroll et al; licensee BioMed Central Ltd. This is an Open Access article distributed under the terms of the Creative Commons
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any medium, pro vided the original work is properly cited.
strengths and limitations of a pragmatic best fit
approach using an existing conceptual model as a start-
ing point to identify a priori themes.
This qualitative evidence synthesis was originally
designed to complement a systematic review and eco-
nomic evaluation on the prevention of colorectal cancer
by reviewing evidence relating both to the attitudes of
adults concerning the taking of named chemopreventive
agents and factors that may inform the related, per-
ceived risk-benefit balance [8]. The agents of interest
were non-steroidal anti-inflammatory drugs (NSAIDs,
including aspirin), vitamins, minerals, folic acid or folate,
selenium, calcium and dietary supplements generally. No
previous evidence synthesis was identified regarding
peoples views about taking these agents, especially for
primary prevention of colorectal cancer. The effective-
ness of any agent is moderated by levels of compliance
with the proposed regimes. For those contemplating tak-
ing such agents, for example to protect against cancer,
the decision-making process can be seen as complex,
due to the uncertainty of the trade-offbetween efficacy
of the agent, i.e. the likelihood of getting the cancer, and
its possible long-term side effects [9]. It has also been
pointed out that people may find it difficult to incorpo-
rate a regular pattern of chemoprevention into the
demands of day-to-day life. On the other hand research
points to the successful use of low-dose aspirin in redu-
cing the risk of heart attack and stroke [10].
The aim of the current paper is to summarise key
results of this synthesis of qualitative studies within the
context of describing the application of a best fit
method, and to consider the lessons learned from adopt-
ing such an approach to framework synthesis.
Methods
Search methods
The aim of the qualitative evidence synthesis was to
examine peoples attitudes towards the taking of agents
or supplements that may be used in the primary preven-
tion of colorectal cancer, i.e. NSAIDs (including aspirin),
vitamins, minerals, folic acid or folate, selenium, calcium
and dietary supplements generally. The synthesis
included studies that focused on exploring the views,
beliefs or attitudes of people who took any of these
agents for any purpose. A systematic search to identify
relevant studies was performed by an information specia-
list following piloting of appropriate search strategies.
The search combined terms describing the agents of
interest (NSAIDs, aspirin, vitamins, etc.) with a pub-
lished, validated filter for identifying qualitative studies,
together with the medical subject heading qualitative
research[11]. The full search strategy is available in the
Appendix. Databases searched for published and unpub-
lished material included MEDLINE, PreMEDLINE,
CINAHL,EMBASE,AMED,ASSIA,IBSS,PsycINFO,
Science Citation Index, and Social Science Citation
Index, and the HMIC and Kings Fund databases. Studies
were limited to those in English published from 2003
onwards to capture contemporary views and attitudes.
Searches were undertaken in June 2008. Given the
problems with identifying social science or qualitative
literature through systematic searching of electronic
databases alone [12,13], the reference lists of all included
studies were checked for additional literature, and a
berry-picking approachutilising supplementary, non-
systematic searching [14] testing various combinations of
terms was also performed by two of the authors (AB,
KC). This iterative, pragmatic approach to searching
aimed to identify a set of studies providing relevant infor-
mation on views and attitudes towards the taking of
potential chemopreventive agents.
Study selection
To be included in the review, a study had to focus on
exploring the attitudes, perceptions and beliefs of adults
(any country) surrounding the taking of the agents listed
above, through qualitative data from interviews or focus
groups, and cross-sectional data from satisfaction sur-
veys,i.e.unstructuredandstructured, but often textual
data describing peoples own, personal, subjective
experiences, views or attitudes relating to the interven-
tion of interest. Previous reviews have also adopted this
inclusive approach to viewsstudies, i.e. including qua-
litative data describing peoples attitudes and beliefs
from satisfaction surveys as well as more traditional
qualitative study designs [6,15]. The authors each
screened a third of the citations for relevance (based on
the inclusion criteria) and references for potential inclu-
sion were discussed within the team. Disagreements or
uncertain inclusions were resolved by discussion or by
retrieval of the full paper to make a definitive judgment.
Full papers of all potentially relevant citations were
screened using the same process. Data from the
included studies were extracted by two of the authors
(CC, KC) using a review-specific form developed follow-
ing piloting on one included paper.
Best fitapproach to framework synthesis
The authors chose the framework synthesis approach
because a published model was identified from the lit-
erature that conceptualised attitudes of adult women to
the taking of vitamins and minerals [16]. The approach
therefore was augmentative and deductive (building on
this existing model or framework), rather than grounded
or inductive (starting with a completely blank sheet).
The model identified did not entirely match the topic
under study, but it was a best-fitand provided a rele-
vant pre-existing framework and themes against which
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to map and code the data from the studies identified for
this review. A list of themes was derived from this
model (see Figure 1) and provided the aprioriframe-
work of themes against which to code the data extracted
from the included studies.
Data for analysis consisted either of verbatim quotations
from study participants or findings reported by authors
that were clearly supported by study data, for example,
four of the five interviewees reported that the views
of family and friends affected their decision-makingor
StagesThemes
Perceivedneed
DecisionͲmaking
Access
Use
1.Familyfactorsaffectingperceivedneed
2.Personalfactorsaffectingperceivedneed
3.Mediarepresentationsofperceivedneed
4.Spendingcapacity
5.MediainputintodecisionͲmaking
6.PhysiciansinputintodecisionͲmaking
7.FamilymembersinputintodecisionͲmaking
8.CommunityinputintodecisionͲmaking
9.PharmacyinputintodecisionͲmaking
10.Access:obtainingmicroͲnutrients
11.Perceivedbenefits
12.Perceivedrisks(negativefactors)
13.Habitualuse
14.Intermittentuse
Figure 1 A priori themes reflecting peoples views about taking potential chemopreventive agents, derived from Huffman 2002[16].
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75% of respondents said that they were concerned about
side effects of NSAIDs. These data were extracted from
the Resultssections of included studies only, as it was
felt that the Discussion and Conclusion sections would
not present any new data, only additional interpretation or
contextualisation of a studysfindings. Two of the authors
(CC, KC) each extracted data from half of the included
studies. Where any relevant data from the included studies
did not translate into any pre-existing themes, a method
was required to capture these data for the analysis. The
published descriptions of framework synthesis do not spe-
cify a particular method for this,sotheauthorsapplied
secondary thematic analysis, an interpretive, inductive
approach grounded in the data based on methods from
primary research, whereby additional themes were created
as needed based on the study data [17]. In this way, the
existing model acted as the basis for the synthesis and
could be built-upon, expanded upon, reduced or added to
by these new data. Each reviewer checked and examined
critically the extraction and categorisation or coding of
data performed by the other. The principal aim of this
process was to examine the first reviewers categorisation
of the data, i.e. either to verify the coding or to challenge it
by offering an alternative.
The authors then discussed the data and resulting
themes, both those from the pre-existing model and
those generated by the novel, inductive thematic analysis
of the extracted study data. A consensus was reached on
which a priori themes were supported by the data, and
whether new themes identified by the reviewers did
actually map either to a pre-existing theme or to one
another (c.p. reciprocal translation [2]). The result was a
finalised list of themes. The primary reviewer (CC) then
offered an interpretation of the relationships between
the themes based in part on the relationships as they
were represented in the original model (see Figure 1),
and also based on the data itself, which suggested, for
example, that the mediainputted into the central pro-
cedural themes of both perceived need and decision-
making. The new model was then critically considered
by all reviewers. A revised conceptual model was there-
fore developed building on the earlier, identified model,
to describe and explain peoples views around the taking
of potential chemopreventive agents.
Consideration of study quality
Published descriptions of framework synthesis typically
exclude studies of lower quality. However this was not
the approach used in this case, representing a further
innovative deviation from the published method [2]. All
studies that satisfied the relevance criteria were included
because there is an increasingly strong case for not
excluding qualitative data studies from evidence synth-
esis based on quality assessment [1,18,19]. Studies were
assessed using key quality criteria derived from relevant
critical appraisal checklists for qualitative studies [20]
and other systematic reviews of peoples views [1,2].
These elements also appear in recent guidance from the
Cochrane Qualitative Research Methods Group [21].
The assessment consisted of querying whether the fol-
lowing are clearly and adequately described in the publi-
cation: the question and study design; how the
participants were recruited or selected; and the methods
of data collection and analysis used (See Additional file
1). The better-reportedstudies provided details on two
or more criteria, whereas the inadequately-reported
studies clearly described no more than one. The deci-
sion only to focus on these four elements, and what was
reported or clearly described by the included studies,
was taken for two reasons. Firstly, these elements of the
study were potentially more easily judged and appre-
hended than others, as they were either described or
not. Secondly, it has been pointed-out previously that
any appraisal checklist is only assessing what has been
reported in a publication [22]. The focus therefore was
on the reporting of basic methods and not potentially
subjective judgements regarding studiesvalidity or relia-
bility [18].
While it is acknowledged that there is always uncer-
tainty concerning how well or poorly a study has been
conducted, if authors clearly describe their approach
and sampling, and data collection and analysis meth-
ods, then this potentially lends greater robustness to
the studys findings. This is because any inherent risk
of biasmay be better determined than if this informa-
tion was absent, regardless of the studys findings. This
does not preclude the possibility that an inadequately-
reportedstudy has actually been well-conducted, but
it does form a reasonable basis for making a quality
assessment. This relatively small number of easily-
defined criteria can also be seen to apply to qualitative
studies universally and may be more practical than
checklists with much larger numbers of questions,
especially as these have been found to generate low
inter-rater reliability scores among otherwise experi-
enced qualitative systematic reviewers [18]. This was
one of the first practical attempts to utilise assessment
criteria based specifically and exclusively on the
description or reporting of a studysmethodandsam-
pling strategies, and methods of data collection and
analysis. No study was excluded on the basis of the
adequacy of its reported processes, but the assessment
aimed to explore quality of reporting as an explanation
for differences in the results of otherwise similar stu-
dies, and to consider its impact on the internal validity
of the review [23]. A sensitivity analysis would be per-
formed in the event of the inclusion of inadequately-
reportedstudies.
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Results
Quantity and quality of included studies
The literature search identified 1,805 unique citations,
15 of which satisfied the inclusion criteria. Five further
studies were identified by the berry pickingapproach
described above [8]. In total, twenty studies were
included. No study failed to describe clearly at least two
of the following: the question and study design, and the
methods of sampling, data collection or analysis. Study
quality, in terms of how well or how poorly studies were
described, was therefore not a potential moderator of
the findings; a sensitivity analysis was not performed.
Data synthesis and development of model
A combination of coding against pre-existing themes and
the generation of and assignment of data to new, agreed
themes, generated the model presented in Figure 2. A full
description of the evidence supporting this model is pub-
lished elsewhere [8]. The model describes the processes
involved in an individuals decision about whether or not
to take possible chemopreventive agents. The process
runs from the first stages of perceived need, on the left,
through the decision-making process itself, to final non-
use or use, and maintenance of use, on the right. External
agents, such as health professionals and family members,
and internal factors, such as a persons own experience
or health, were all found to impact both on an indivi-
duals perceived need for an agent or supplement, as well
as their subsequent decision about whether or not to
take it.
Usefulness of the preliminary conceptual framework in
assigning data to themes
Since the source of the preliminary framework was a
single published model, the manner in which new
themes built-on, developed and altered this preliminary
conceptual framework is quite transparent. In this
review, this may be assessed in part by comparing
Figure 1 with Figure 2. The principal procedural ele-
ments of the preliminary model also held true for this
sample of studies and their population, i.e. the transition
through the stages of perceived need, decision-making,
risk versus benefit and use or non-use. These elements
also reflect the three key stages of Contemplation,
Determination and Action in Prochaska and Velicers
model (1997) of the development of health behaviours,
which was later found to be relevant [24]. The a priori
identification of these key constructs therefore enabled
Externalfactors
Internal/Personalfactors
ACTION
DETERMINATION
CONTEMPLATION
DecisionͲmakingPerceivedneedRisk/Benefit
Balance
Useand
maintenance
Benefit
Risk
Physical
p
ro
p
erties
Age andgender
Sel
f
Ͳefficacy
Health status
Observation/
ex
p
erience
SocioͲ
demo
g
ra
p
hic
Pharmacy
OtherpeopleDoctors
Cost
Family
Media
CredibilityandClarity
Perception
Perception
Experience
Experience
Figure 2 Conceptual model to describe views and experiences of adults concerning the taking of potential chemoprevention agents.
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therapidcodingofstudydatafromthisreviewagainst
these tested and highly relevant components of health
behaviour decision-making. The preliminary framework
also provided themesthat informed the perceived
needand decision-makingstages of the model (see
numbers 1-9 in Figure 1). Once clear definitions had
been applied to each of these themes, the study data
were coded rapidly against them. Very little study data
were coded against the themes of Spending capacity
(or Costs)andAccess: obtaining the agent,which
may reflect differences in the cultural context of the
preliminary conceptual model (a low-income country in
South America) compared with the studies included in
the review (principally UK, Europe and North America).
However, relatively more substantial amounts of data
were coded against the remaining themes.
Extension of the preliminary conceptual framework to
generate the final model
Despite these helpful overlaps, which permitted rapid
and reliable coding of much data from the included stu-
dies, the preliminary model lacked sufficient depth or
complexity to explain all thedataintheincludedstu-
dies. As with the preliminary model, some factors influ-
enced both need and decision-making. For example, the
influences of family and the media were present at both
of these stages, but the categorisation of these factors
was re-specified in the new model. Family, media, physi-
cians, other people and pharmacy were all designated in
the new model as external factors having input into per-
ceived need and decision-making. The personal factors
theme from the original model was re-specified as
internal or personal factorsto include an individuals
own observations or experience, their health and socio-
economic status, age and gender, and their sense of self-
efficacy. All of these characteristics were found in the
included studies to affect perceived need and decision-
making. It was felt that the pre-existing theme of per-
sonal factorsalone was insufficient to illustrate the
complexity of factors at play. The role of age, gender
and the physical properties of agents were new factors
identified by the synthesis affecting the a priori theme
of use, which were absent from the original conceptual
model.
Relationships between the themes were not well-devel-
oped in the preliminary model. The synthesis found that
not only did family, physicians and others affect decision
making, but also that this relationship was moderated by
the credibility of the source and the clarity of the infor-
mation being given. Perceived risks and benefits were
key pre-existing themes shaping use, but the moderating
role of personal experience was an additional element
identified by the synthesis for the new model. Further-
more the risk/benefit balance theme was also found to
have an ongoing, potentially recursive influence on deci-
sion-making and agent use. Indeed, unlike the existing
models, which appear to be exclusively linear, the model
that resulted from this synthesis was potentially more
recursive: the decision-making stage might still be revis-
ited on the basis of side-effects ("risks) experienced at
the stage of use. This new model can therefore be seen
not only to validate, but also to build upon, extend and
contextualise existing, relevant published models. The
aprioriboxes of Contemplation, i.e. perceived need;
Determination, i.e. decision-making; and Action, i.e. use
and maintenance, have been opened to reveal the com-
plexities of the factors therein, their relationships and
moderators.
Discussion
The model generated by the framework synthesis
describes the processes involved in an individuals deci-
sion about whether to initiate and keep taking potential
chemopreventive agents. External agents, such as health
professionals and family members, and internal factors,
such as a persons own experience or health status,
combine to impact on an individuals perceived need for
an agent or supplement, and their subsequent decision
about whether or not to take it. Decision-making was
strongly influenced by perceived risks and benefits asso-
ciated with an agent or supplement. Firstly, perceived
risks and benefits directly influence an individualsdeci-
sion to take an agent. Secondly, they may inform a per-
sonal assessment of the trade-off between risk and
benefit, thus affecting the decision-making process. It
has been reported elsewhere that decision-making
regarding agents for chemoprevention or symptom man-
agement may be affected both by health status, for
example, a cancer diagnosis [25,26], and by peoples per-
ceived need for an agent and perceived risks associated
with that agent [27-29]. The model generated by this
review highlights the complex influences at work in this
decision-making process.
This review applied a form of framework synthesis to
analyse the data, based on a single best fitmodel iden-
tified in the literature. This approach differs from other
published versions of framework synthesis in which the
aprioriframework was developed from a range of
sources, including familiarisation with and consultation
around the published background literature, both theo-
retical and empirical, and personal experiences [5,6].
The approach taken here is of potential value for sys-
tematic reviewers as it does not require such extensive
literature review, consultations or topic expertise to
develop an a priori framework before embarking on the
review itself. This may be of particular value when
undertaking a synthesis of qualitative evidence within
the limited timeframes of a health technology
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assessment, for example. Projects such as Health Tech-
nology Assessments, produced in multidisciplinary cen-
tres with contractual obligations, with a six-month or
one-year span, and which also involve reviews of effec-
tiveness, cost-effectiveness, mathematical modelling and,
in some cases, qualitative evidence synthesis, often pre-
sent challenges in relation to timeliness and the avail-
ability and expertise of members of research teams [30].
In this particular case study, the qualitative evidence
synthesis was conducted after the effectiveness synthesis,
which required the qualitative synthesis to be fairly
quick within the projects required timeframe. However,
a temporal dependency between the two types of synth-
esis will not always exist, and so a more in-depth quali-
tative approach may be possible for some projects.
However, if a framework of related, relevant concepts
already exists, then the approach used here permits a far
more rapid identification of the aprioriframework; it
also permits more rapid and structured coding and
synthesis of data from the reviews included studies than
grounded-theory techniques. In this way, where existing
theories or models exist, they can be tested against the
evidence for the reviews own particular criteria and evi-
dence. This approach is therefore potentially more prag-
matic than other forms of qualitative data synthesis. The
identification and use of a model that was overtly best
fit, and therefore carried shared acknowledgment
within the team that it was contingent on emerging data
also empowered the reviewers to resist the inclination to
slotstudy findings into a generic framework. This
potentially enabled individual team members to privilege
context-specific insights that emerged from this review
over the generic observations already present within the
pre-existing model. Furthermore it provided a mechan-
ism for flagging up and explicitly communicating diver-
gent findings or themes within the review team. The
resultant synthetic product is expressed as an enhanced
model recording each key dimension identified; the nat-
ure of the concepts under study; and associations
between themes and tensions between them [6].
The method is however dependent on the identifica-
tion of an appropriate existing conceptual model. The
review team sought to identify such a model by combin-
ing a sensitive string of search terms (e.g. model$ OR
framework$ OR theoretical OR theory OR concept OR
conceptual) with terms representing the health-related
behaviour of interest. This approach was employed
firstly on a bibliographic database (PubMed MEDLINE)
but was found to be limited by poor coverage of theore-
tical aspects in published abstracts. A more productive
approach proved to be using Google Scholar with the
same string of search terms, and certainly the potential
for this approach to be used with other collections of
full-text documents remains to be further explored. This
strategy was conceived as iterative and purposive: it
required search strategies that aimed to maximise the
likelihood of retrieving a model of pragmatic utility to
the project; the aim was not the systematic identification
of all such models.
Furthermore the approach used for this particular case
study was predicated on the review teams belief that the
key criterion of the appropriateness of such a model
most likely related to the health-related behaviour of
interest, i.e. attitudes to the long-term taking of particu-
lar dietary supplements or similar agents. The popula-
tion and the agents themselves may be less critical in
such cases, although the closer the fit to the population
and intervention of interest, the better. This is why we
describe it as a best-fitapproach. In this case study,
young women and vitamins or micro-nutrients formed a
sub-set of the populations and agents of interest. The
conceptual model therefore had limited external validity
but was still externally valid.
Some issues were encountered when piloting this best
fitframework synthesis method. When initially seeking
to code the extracted data from the included studies
using the themes derived from the relevant model, the
two reviewers were not always coding the same data
against the same themes. It therefore became apparent
that each of the apriorithemes had to be clearly
defined in order to facilitate the coding process. The
subsequent provision of clear consensual definitions not
only enhanced the reliability of the coding, but also
strengthened the rigour of the synthesis. It should be
recognised, however, that while consensus between
reviewers strengthens internal validity this does not
necessarily ensure congruence with the original mean-
ings intended by the author of the framework (external
validity). In this sense a form of reciprocal translation
is taking place but via use of a conceptually rich index
paper(many-to-one), rather than across all included
studies (many-to-many), as intended by the originators
of meta-ethnography [31]. Such considerations have
been neither identified nor articulated in previous
studies.
It further became apparent that additional analysis was
needed to interpret and analyse data which could not be
reliably assigned to any of the pre-existing, apriori
themes, or, in the case of personal factors,forwhich
the pre-existing theme was inadequate. In this sense the
usefulness of a particular framework is not only deter-
mined by conceptual fitbut also by pragmatic con-
cerns of what proportion of the study data can be
accommodated within it. Further thematic analysis of
data from the included studies was therefore required.
This was completed by the first author using standard
thematic analysis techniques, and the results examined
critically by the other two reviewers. The resulting,
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agreed new themes were then incorporated with the
pre-existing themes into a new conceptual model that
captured the data and reflected a possible network of
relationships between those data-driven themes. The
existing published descriptions of the framework synth-
esis method do not detail particular techniques for ana-
lysing data that are not captured by the preliminary
framework, how any such new themes are to be incor-
porated into the final model, or how the relationships
between these themes may be expressed.
Finally, this review did not exclude studies on the
basis of quality, thereby deviating from one element of
the published description of framework synthesis [2].
The internal validity of a review depends in part on the
quality of included studies and the reliability of their
findings. Currently there is much debate and little con-
sensus around the feasibility and usefulness of quality
assessments of qualitative studies in evidence synthesis
[18]. Some techniques, such as meta-ethnography [4],
and the previously published form of framework synth-
esis, actively exclude studies on the basis of the quality
assessment. The quality assessment for this review
focused on reporting of study design, sampling strategies
and methods used for data collection and analysis.
These items were the most frequently reported and
easily apprehended elements of study design. They thus
offered a reasonable route for identification of potential
risk of bias. All twenty included studies were assessed as
being of similar, generally satisfactory quality, so, from
this perspective, study quality did not provide a poten-
tial explanation for any differences in findings. The issue
of the inclusion or exclusion of studies for this type of
synthesis, based on their assessed quality, therefore
remains unresolved based on this case study.
Methodologically the authors found this best fit
approach to framework synthesis, as developed and
tested in this review, to be a useful, fairly rapid and reli-
able and, above all, pragmatic method of synthesising
qualitative data. This best fitapproach to synthesis
was therefore found to work well overall, particularly
within the role previously identified as an existing
strength, namely for testing existing potentially generali-
sable theories and models within a specific context.
However, such a best fitapproach would benefit from
further testing and refinement.
Limitations
Thisisasinglecasestudyevaluatingtheapproach
described; additional studies testing this approach to
qualitative evidence synthesis need to be undertaken.
Also, as an approach, it is only viable if an appropriate
model already exists in the literature. The other pub-
lished models for framework synthesis circumvent this
problem as the a priori framework is generated by the
research team itself. It is also the case that an apparently
appropriate a priori model may be found only to accom-
modate a small proportion of the data from a reviews
included studies. In such a case, secondary thematic
analysis would form the principal approach to synthesis,
thus reducing the major potential pragmatic benefits of
the best-fit approach described in this paper. Reviewers
must therefore exercise careful consideration of the
potential external validity of existing models based on
the behaviour and population of interest.
Conclusion
This best fitmethod of framework synthesis utilised
current methodological developments within qualitative
data synthesis for systematic review and the production
of accompanying conceptual models and frameworks.
The case study was a systematic review of adultsviews
about taking various potential chemopreventive agents.
The best fitframework synthesis offered a means to
reinforce, critique and develop an existing published
model, conceived for a different but relevant population.
Being able to start from apriorithemes, rather than
generating theory grounded in data, produced a rela-
tively rapid process when compared to more interpreta-
tive forms of synthesis. However this best fitmethod
still requires analysis of data that are not captured by
the preliminary model. The authors suggest that this
best fitapproach occupies a pragmatic middle ground
between grounded theory-type and framework based
syntheses and acknowledge the need for further
evaluation.
Appendix
Database: CINAHL - Cumulative Index to Nursing &
Allied Health Literature
Search Strategy:
1 vitamin$.tw.
2 mineral$.tw.
3 folate$.tw.
4 selenium.tw.
5 calcium.tw.
6 exp Dietary Supplements/
7 Dietary Supplementation/
8 dietary supplement$.tw.
9 non-steroidal$.tw.
10 non steroidal$.tw.
11 nonsteroidal$.tw.
12 NSAID$.tw.
13 antiinflammator$.tw.
14 anti-inflammator$.tw.
15 anti inflammator$.tw.
16 aspirin$.tw.
17 or/1-16
18 interview$.tw.
Carroll et al.BMC Medical Research Methodology 2011, 11:29
http://www.biomedcentral.com/1471-2288/11/29
Page 8 of 9
19 experience$.tw.
20 qualitative$.tw.
21 exp Qualitative Studies/
22 or/18-21
23 17 and 22
24 limit 23 to yr="2003 - 2008
Additional material
Additional file 1: The question and study design; how the
participants were recruited or selected; and the methods of data
collection and analysis used.
Acknowledgements
The case study on which this work was based was part of a larger project
funded by the UK NCCHTA (06/70/01)
Authorscontributions
CC and AB conceived the study; CC designed the study; CC, KC and AB
extracted the data and appraised included studies; CC, KC, and AB analysed
and interpreted the data. CC drafted the paper and KC and AB undertook
critical revision of important content of the manuscript. All authors approved
the final version of the manuscript.
Competing interests
The authors declare that they have no competing interests.
Received: 13 December 2010 Accepted: 16 March 2011
Published: 16 March 2011
References
1. Thomas J, Harden A: Methods for the thematic synthesis of qualitative
research in systematic reviews. BMC Medical Research Methodology 2008, 8.
2. Barnett-Page E, Thomas J: Methods for the synthesis of qualitative
research: a critical review. BMC Medical Research Methodology 2009, 9.
3. Pope C, Ziebland S, Mays N: Qualitative research in health care: Analysing
qualitative data. British Medical Journal 2000, 320:114-116.
4. Campbell R, Pound P, Pope C, Britten N, Pill R, Morgan N, et al:Evaluating
meta-ethnography: a synthesis of qualitative research on lay
experiences of diabetes and diabetes care. Social Science & Medicine 2003,
65:671-684.
5. Oliver S, Rees R, Clarke-Jones L, Milne R, Oakley A, Gabbay J, et al:A
multidimensional conceptual framework for analysing public
involvement in health services research. Health Expectations 2008,
11:72-84.
6. Brunton G, Oliver S, Oliver K, Lorenc T: A Synthesis of Research Addressing
Childrens, Young Peoples and ParentsViews of Walking and Cycling
for Transport London. London, EPPI-Centre, Social Science Research Unit,
Institute of Education, University of London; 2006.
7. Nilsen E, Myrhaug H, Johansen M, Oliver S, Oxman A: Methods of
consumer involvement in developing healthcare policy and research,
clinical practice guidelines and patient information material. Cochrane
Database of Systematic Reviews 2006, , 3: CD00456.
8. Cooper K, Squires H, Carroll C, Papaioannou D, Booth A, Logan R, Hind D,
Tappenden P: Chemoprevention of colorectal cancer: systematic review
and economic evaluation. Health Technology Assessment 2010, 14:32.
9. Heisey R, Pimlott N, Clemons M, Cummings S, Drummond N: Womens
views on chemoprevention of breast cancer: qualitative study. Canadian
Family Physician 2006, 52:624-625.
10. Lynch P: The role of aspirin in the prevention of polyp recurrence: What
is the right dose? Current Colorectal Cancer Reports 2007, 3:24-28.
11. Wilczynski N, Marks S, Haynes R: Search Strategies for Identifying
Qualitative Studies in CINAHL. Qualitative Health Research 2007,
17:705-710.
12. Papaioannou D, Carroll C, Booth A, Sutton A, Wong R: Literature searching
for social science systematic reviews: consideration of a range of search
techniques. Health Information and Libraries Journal 2010, 27:114-122.
13. Grayson L, Gomersall A: A difficult business: finding the evidence for
social science reviews. ESRC UK Centre for Evidence Based Policy and
Practice. Working paper 19 2003.
14. Sandelowski M, Barroso J: Searching for and Retrieving Qualitative
Research Reports. In Handbook for synthesizing qualitative research. Edited
by: Sandelowski M, Barroso J. New York, NY: Springer; 2007:35-74.
15. Thomas J, Harden A, Oakley A, Oliver S, Sutcliffe K, Rees R: Integrating
qualitative research with trials in systematic reviews. British Medical
Journal 2005, 328:1010-1012.
16. Huffman S: Can Marketing Of Multiple Vitamin/Mineral Supplements
Reach the Poor? The Vitaldía Project, Bolivia; 2002 [http://www.phishare.
org/files/338_Can%20Marleting...Vitaldia.pdf].
17. Miles M, Huberman A: Qualitative Data Analysis: A Sourcebook of New
Methods. 2 edition. Newbury Park, CA: Sage; 1994.
18. Dixon-Woods M, Sutton A, Shaw R, Miller T, Smith J, Young B, et al:
Appraising qualitative research for inclusion in systematic reviews: a
quantitative and qualitative comparison of three methods. Journal of
Health Services Research and Policy 2007, 12:42-47.
19. Dixon-Woods M, Bonas S, Booth A, Jones D, Miller T, Sutton A, et al:How
can systematic reviews incorporate qualitative research? A critical
perspective. Qualitative Research 2006, 6:27-44.
20. Tong A, Sainsbury P, Craig J: Consolidated criteria for reporting
qualitative research (COREQ): a 32-item checklist for interviews and
focus groups. International Journal for Quality in Health Care 2007,
19:349-357.
21. Hannes K: Critical appraisal of qualitative research. Cochrane Qualitative
Research Methods Group Handbook 2010.
22. Dixon-Woods M, Shaw R, Agarwal S, Smith J: The problem of appraising
qualitative research. Quality and Safety in Health Care 2004, 13:223-225.
23. Golafshani N: Understanding Reliability and Validity in Qualitative
Research. The Qualitative Report 2003, 8:597-607.
24. Prochaska J, Velicer W: The transtheoretical model of health behavior
change. American Journal of Health Promotion 1997, 12:38-48.
25. Demark-Wahnefried W, Peterson B, McBride C, Lipkus I, Clipp E: Current
health behaviors and readiness to pursue life-style changes among men
and women diagnosed with early stage prostate and breast carcinomas.
Cancer 2000, 88:674-684.
26. Strecher V, Rosenstock I: The health belief model. In Health Behavior and
Health Education. Edited by: Rimer BK. San Francisco: Jossey-Bass;
1997:41-59.
27. Floyd D, Prentice-Dunn S, Rogers R: A meta-analysis of research on
protection motivation theory. Journal of Applied Social Psychology 2000,
30:407-429.
28. Milne S, Sheeran P, Orbell S: Prediction and intervention in health-related
behavior: a meta-analytic review of protection motivation theory. Journal
of Applied Social Psychology 2000, 30:106-143.
29. Horne R, Weinman J: Self-regulation and self-management in asthma:
Exploring the role of illness perceptions and treatment beliefs in
explaining non-adherence to preventer medication. Psychology & Health
2002, 17:17-32.
30. Rotstein D, Laupacis A: Differences between systematic reviews and
health technology assessments: a trade-off between the ideals of
scientific rigor and the realities of policy making. International Journal of
Technology Assessment in Health Care 2004, 20:177-183.
31. Noblit GW, Hare R: Meta-ethnography: synthesizing qualitative studies
Newbury Park, CA: Sage; 1998.
Pre-publication history
The pre-publication history for this paper can be accessed here:
http://www.biomedcentral.com/1471-2288/11/29/prepub
doi:10.1186/1471-2288-11-29
Cite this article as: Carroll et al.: A worked example of best fit
framework synthesis: A systematic review of views concerning the
taking of some potential chemopreventive agents. BMC Medical Research
Methodology 2011 11:29.
Carroll et al.BMC Medical Research Methodology 2011, 11:29
http://www.biomedcentral.com/1471-2288/11/29
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