Family resilience and adaptive coping in children with juvenile idiopathic arthritis: protocol for a systematic review
Saetes et al. Systematic Reviews
Family resilience and adaptive coping in children with juvenile idiopathic arthritis: protocol for a systematic review
Sophia Saetes 0 1
Lisa Hynes 1 3
Brian E. McGuire 0 1
Line Caes 1 2
0 School of Psychology & Centre for Pain Research, National University of Ireland , Galway , Ireland
1 Footnotes Twitter: Follow Sophia Saetes at @SophiaChrissa
2 School of Natural Sciences, University of Stirling, Stirling , Scotland , UK
3 Department of Psychology, West Virginia University , Morgantown, WV , USA
Background: This systematic review is the first step in a study investigating the resilience methods and processes in families of children with juvenile idiopathic arthritis. In particular, this review will focus on chronic or persistent pain, as a common symptom of juvenile idiopathic arthritis, which is the most common rheumatic disease in childhood. The experience of persistent pain can add to the functional disability associated with juvenile idiopathic arthritis. Resilience has relevance to all areas of paediatric psychology, and targeted attention to child, sibling, and parent strengths within the context of paediatric chronic pain and juvenile idiopathic arthritis in particular will augment the field on numerous levels. The objective is to determine which resilience processes are associated with a favourable quality of life in terms of academic, communication, emotional, interpersonal, physical, psychological, and social well-being in families of children with chronic pain associated with JIA. Methods/design: This systematic review will be conducted and reported in accordance with the Preferred Reporting Items for Systematic Reviews and Meta-Analyses statement and the PRESS (Peer Review of Electronic Search Strategies) guideline. Longitudinal, cross-sectional, and treatment studies written in English will be included, as will grey literature (i.e. conference abstracts and dissertations). Studies involving participants who are 6-18 years of age, have been diagnosed with juvenile idiopathic arthritis, are experiencing chronic pain, and are currently undergoing treatment will be included regardless of sex, arthritis type, and type of treatment. Studies including siblings who are 6-18 years of age and the patient's parents will be included. Discussion: Research exploring resilience within the adult population is accruing. Shifting our focus to protective factors of resilience in the context of paediatric chronic pain, specifically juvenile idiopathic arthritis, is a novel and promising pursuit that has the potential to optimize and inform future clinical practice and interventions. A better understanding of the role of reliance in family adaptation will facilitate the development of more effective treatment approaches and lay the foundation for more effective self-management in paediatric chronic pain. Systematic review registration: This protocol is registered with the International Prospective Register of Systematic Reviews (PROSPERO) database (registration: CRD42016047226).
Description of the condition
This systematic review is the first step in a larger study
investigating the resilience methods and processes in
families of children with juvenile idiopathic arthritis
(JIA). In particular, this review will focus on chronic or
persistent pain, a common symptom of JIA, which is the
most common rheumatic disease in childhood.
Symptoms of JIA include joint pain and stiffness, fevers, rash,
bone infection, and inflammation. JIA frequently
continues into adulthood and can end in substantial
longterm morbidity, including physical disability [
experience of persistent pain can add to the functional
disability associated with JIA.
Why is resilience important?
In the context of paediatric traumatic chronic illness
such as JIA, resilience may be a one process that
determines whether the child and their family experience
traumatic stress or post-traumatic growth. Resilience is
considered a protective factor and can be defined as “a
dynamic and multi-systemic progression that allows the
individual to respond effectively when faced with risk or
adversity (e.g. medical condition)” [
]. The process of
resilience originates within the individual and is enhanced
through developmental, social, cultural, and
environmental factors. Resilience encompasses the concepts of
posttraumatic growth, benefit-finding, optimism, and hope.
Only a few studies have focused on these concepts in
children and adolescents outside the context of childhood
cancer, but preliminary evidence suggests that these
constructs may be related to good adjustment in various
paediatric chronic illnesses [
]. Current practices
pertaining to paediatric chronic pain are guided largely by an
adult-focused body of research, which is unsuccessful in
taking into account relevant developmental factors such
as cognitive and social development and complex familial
]. Further research into the presence, assessment,
and utilization of resilience within paediatric chronic pain
Resilience has relevance to all areas of pediatric
psychology, and targeted attention to child, sibling, and family
strengths within the context of paediatric chronic pain
and JIA in particular will augment the field on numerous
]. For instance, by refining the particular
strengths or resilience resources that are of relevance for
adaptive adjustment to childhood JIA, these processes
can be targeted in clinical practice in order to enhance
psychological, cognitive, and social outcomes for families
of children with JIA.
While there are several models emerging in the field
that aid in the measurement of resilience, these models
require more research on how they may apply to
resilience theory. Furthermore, though resilience theory
includes risk exposure within adolescents, it primarily
concentrates on healthy growth despite risk exposure.
As such, the authors chose to engage in a broad
approach (the inclusion of both resilience resources and
mechanisms) for this systematic review.
The objective of this study is to determine which
resilience processes are associated with a favourable quality
of life, in terms of academic, communication, emotional,
interpersonal, physical, psychological, and social
wellbeing, in families of children with chronic pain
associated with JIA.
Criteria for considering studies for this review
This systematic review, which will result in a narrative
synthesis of the current evidence on family resilience in
JIA, will be conducted and reported in accordance with
the Preferred Reporting Items for Systematic Reviews
and Meta- Analyses (PRISMA) statement and the PRESS
(Peer Review of Electronic Search Strategies) guideline.
The PRISMA-P+ checklist document shows this in more
detail [see Additional file 1] [
]. The PRISMA-P
checklist encourages authors to describe eligibility
criteria using the PICO reporting system (which describes
the participants, interventions, comparisons, outcome(s),
and study design of the included studies) . This
systematic review protocol is registered with the International
Prospective Register of Systematic Reviews (PROSPERO)
database (registration number: CRD42016047226).
Types of studies
Longitudinal, cross-sectional, and treatment studies
written in English will be included. There were no limitations
with respect to the design of treatment studies to be
eligible for inclusion. Grey literature (i.e. conference
abstracts and dissertations) will be included. Review articles
will be excluded from the search, as will studies for which
the full text is not available.
Types of participants
Studies involving young people who are 6–18 years of
age, who have been diagnosed with JIA, who are
experiencing chronic pain, and who are currently undergoing
treatment will be included regardless of sex, arthritis
type, and type of treatment. Studies including siblings
who are 6–18 years of age and the patient’s parents will
also be included.
Types of outcome measures
Included studies must have a variable reflecting
resilience as either the primary outcome of interest or as a
key predictor, moderator, or mediator. This will include
resilience utilized as a resource or a mechanism.
Resilience resources can include optimism, social and
community support, self-esteem, problem-solving/active
coping, positive family/social interactions, and adaptive
family functioning [
]. Resilience mechanisms identified
specifically for the context of pain are pain acceptance,
pain-related self-efficacy, parental acceptance of pain,
and parent psychological flexibility [
]. Resilience has
been operationalized by several terms, including but not
limited to post traumatic growth or benefit seeking,
adjustment or adaptation, self-esteem, self-concept,
optimism, and hope, all of which will be searched for.
Studies evaluating the impact of resilience variables, either
direct or indirect, on children with JIA and their families
in terms of impact on quality of life and functional
outcomes (academic, communication, emotional,
interpersonal, physical, psychological, and social) will be included.
Search methods for identification of studies
No date restriction will be imposed on the studies.
Studies will be included if a full-text paper in English is
available, either through databases or through contact with
the study authors. Where available, protocol methods
will be compared with the methods and results reported
in the included study.
The following electronic databases will be searched: Web
of Science, MEDLINE (Ovid), EMBASE, Psychology and
Behavioral Sciences Collection (EBSCO), Psycarticles
(Ovid), and PsycINFO (Ovid). This can be seen in Table 1.
The same search strategies will be used with alterations as
appropriate for each database interface.
Searching other resources
Unpublished and ongoing trials will be identified by
checking trials and protocols published on relevant
databases of current ongoing clinical research studies (e.g.
https://clinicaltrials.gov). Trial registries will also be
searched. The lead or contact authors of all identified
studies will be asked to identify further studies where
possible. Grey literature will be searched using the Open
Grey database (www.opengrey.eu/), which includes
technical or research reports, doctoral dissertations, and
conference papers from the previous 5 years (e.g. from
International Symposium on Paediatric Pain (ISPP), the
1. (Juvenile Idiopathic Arthritis or JIA or rheumatoid arthritis or
systemiconset Juvenile Idiopathic Arthritis or psoriatic arthritis or
enthesitisrelated Juvenile Idiopathic Arthritis or oligoarthritis or polyarthritis).
2. AND (Chronic pain or recurrent pain or pain).
3. AND (Children or child or adolescence or adolescent or pre
adolescence or pediatric or paediatric).
4. AND (Sibling or family or family function or parent or parenting or
parental or peer relationships).
5. AND (Resilience or resiliency or post traumatic growth or optimism or
benefit seeking or benefit finding or coping skills or coping or
adjustment or adaptation or health behavior or health behaviour or
quality of life or hope or psychological resilience or psychosocial
functioning or social support or self-concept or acceptance or
selfefficacy or positive affect).
Society of Paediatric Psychology Annual Conference
(SPPAC), or the Paediatric Pain Conference (PPC)
conferences). In addition, reference chaining will be
implemented by reviewing the reference lists of all included
papers to identify any significant articles that may have
Data collection and analysis
Selection of studies
One review author (SS) will initially screen titles and
abstracts and eliminate those obviously not relevant to this
review. A second review author (LH) will also screen
20% of all titles and abstracts and full text articles.
Ineligible studies will be excluded at this stage, and the
authors will record the reason for rejection. When the title
and abstract do not provide all the information
concerning the criteria, full-text copies will be retrieved and
screened. The review authors will retrieve full-text copies
of all studies if either review author determines that the
study possibly or definitely meets the inclusion criteria.
Disagreements between the two reviewers will be
resolved by discussion, with the involvement of a third
reviewer where agreement cannot be reached (LC or BMG).
Multiple reports of the same study will be counted as a
single study. The PRISMA template will be used to
produce a flow chart showing details of studies included and
excluded at each stage of the study selection process.
Data extraction and management
A data extraction sheet will be created and piloted on a
sample of three studies and then altered if required before
full data extraction begins. The two review authors will
then extract the data from the selected studies.
Discrepancies will be resolved by discussion, with the involvement
of a third reviewer where necessary. Authors will be
contacted in order to obtain any necessary missing data. The
following information will be extracted from the studies:
Participant characteristics including demographic
characteristics (e.g. age, sex)
Disease-specific factors such as JIA type, longevity,
pain variables, and treatment characteristics
including types and duration of treatment
Sibling and family data (e.g. ages, sex,
The geographic location of the study
Inclusion/exclusion criteria for participation in study
Relevant findings with respect to the level of family
resilience and its impact on child QOL
Classification of the resilience variables as
predictors, moderators, mediators, or outcome
Quality assessment of the included studies
The criteria to be used were developed to assess
study quality for qualitative, quantitative, and mixed
methods studies [
]. These criteria were established
in response to an appeal by various published
11, 12, 13, 14, 15, 16, 17
] to evaluate the
scientific quality of the studies a researcher chooses for a
To apply this method, the researchers (SS & LH) will
follow the guidelines by rating the quantitative studies
on nine criteria, e.g. explicit scientific context and
purpose, methods, measurement reliability and statistics,
statistical power, internal validity, measurement validity,
external validity, appropriate discussion, and
contribution to knowledge. Accordingly, the qualitative studies
will be rated on 11 criteria, e.g. explicit scientific context
and purpose, methods, grounding in examples, the
findings are integrated into a framework, owning one’s
perspective, resonating with readers (the material is
presented in such a way that readers, judge it to have
represented accurately the subject matter or to have clarified
or expanded their appreciation and understanding of it
]), data is appropriate, credibility checks, situating the
sample, appropriate discussion, and contribution to
knowledge. To address studies using mixed methods
(qualitative and quantitative designs), SS and LH will
utilize all the presented criteria. Papers will be rated on
a scale from low to high, where 1 represents low quality
and 3 represents high quality [
]. Each paper will
receive an average score over the various criteria. In both
the qualitative and quantitative criteria decision making
process, disagreements between SS and LH will be
mediated by either BMG or LC.
Two review authors (SS and LH) will systematically and
comprehensively assess the results of each study, explore
the relationships in the data, and highlight the vital
characteristics of the studies where relevant, such as
important similarities or differences (for example, in
study design, populations, interventions or other
elements). Further, the review authors will assess the
robustness of the synthesis based on the amount and
quality of the evidence (i.e. the included studies), and
the methods used to synthesize the evidence (i.e. how
well the methods minimized bias). The results will be
synthesized according to the Resilience Resources and
Mechanisms in Paediatric Chronic Pain model [
The authors will further evaluate the quality of the
studies using the Quality Assessment developed by
Alderfer et al. [
] through which papers are rated on a
scale of 1 (low—little or no evidence of fulfilling the
guideline or doing poorly) to 3 (high—good evidence or
The researchers will be utilizing a text-based analysis
in the form of a narrative synthesis. The researchers will
refer to the Guidance on the Conduct of Narrative
Synthesis in Systematic Reviews [
]. A narrative synthesis
is appropriate in this context as the evidence is varied
and underdeveloped. While a narrative synthesis can
involve a manipulation of the data, we will not be taking
that approach. Instead, the researchers will be focusing
on a wide range of questions and summarizing the
findings across the data. In addition, any measures that
assess QOL and resilience in the studies will be included
in the narrative synthesis.
Subgroup analysis and investigation of heterogeneity
We expect different categories will be used by the
included studies, including context of the chronic pain,
types of resilience processes and mechanisms, and
individual as opposed to family dynamics. Therefore, if
sufficient data are available, we will undertake subgroup
analysis based on the following:
1. Types of resilience: mechanism or resource 2. Individual approaches to resilience processes and mechanisms versus the family approaches to resilience processes and mechanisms
If appropriate, we will use subgroup analysis to
categorize these studies and explore heterogeneity.
The proposed review will add to the literature in several
ways. Research efforts to further the understanding of
paediatric chronic pain experiences are usually focused
on vulnerability factors for maladaptive coping in both
children and their family, such as anxiety [
], pain catastrophizing [
], and fear [
However, developmental psychology research has shown that
many family units demonstrate substantial flexibility and
resilience during negative effects or ongoing interference
due to chronic illness [
Research exploring resilience within the adult
population is rapidly accruing [
], and it supports positive
emotional responses as a resilience resource [
]; however, the
focus on resilience within the context of paediatric
chronic pain is only recent [
]. In particular, there has
been a scarcity of longitudinal approaches investigating
how resilience can promote adjustment in children with a
chronic illness [
]. Shifting our focus to protective factors
and mechanisms of resilience in the context of paediatric
chronic pain, specifically JIA, is a novel and promising
pursuit that has the potential to optimize and inform
future clinical practice and interventions. Recent literature
validates family functioning as having a high level impact
on child outcomes and demonstrates the positive effects
of family-level processes and variables as resilience tools
within paediatric chronic pain. However, this research is
restricted to studies examining general family functioning
and resilience instead of measures specific to chronic pain
or JIA [
]. Though literature regarding family adjustment
after a diagnosis of paediatric chronic illness is available
], this work has not been conceptualized within the
framework of family resilience theory [
research into the resilience of the family system, including
parents and siblings, is important to understand the
impact on the family as a whole, as they often alter roles and
responsibilities to effectively communicate, manage
emotions, and successfully work as a team to meet treatment
As a first important step in this direction, this review
will summarize the current evidence on how resilience
affects the parents and siblings on an individual level
while simultaneously concentrating on how resilience
affects the family unit. This will be achieved through the
application of the Resilience Resources and Mechanisms
in Chronic Pain model developed by Cousins et al. [
the main analytical framework. This model will highlight
and synthesize the findings in an accurate and
constructive format. In addition, the use of the Resilience
Resources and Mechanisms in Chronic Pain model will
strengthen the value and quality of the summary as well
as the rigorousness and thoroughness of the data.
Briefly, the model extrapolates the relationship
between “resilience resources” and “resilience mechanisms”
in the framework of paediatric chronic pain and has
been successfully utilized within paediatric asthma and
diabetes populations. The Resilience Resources model
has identified several intricate systems that promote
health, illness management, adaptability, and
psychosocial functioning [
]. Potentially important additional
constructs to be explored within this area include
positive affect, post-traumatic growth, benefit finding,
optimism, and self-regulation towards pain [
The authors of the Resilience Resources model
acknowledge that both individual and family-based
processes impact paediatric pain functioning and influence
]. Their model was adapted from Sturgeon
and Zautra’s adult chronic pain risk-resilience model;
however, Cousins et al. [
] adjusted it to include
variables that are specific to, and have received support
from, the paediatric chronic pain literature. Cousins et
] stress that while research supports the negative
effects of paediatric chronic pain, there is limited research
available on positive adaptation and the extent to which
resilience influences the family as a unit.
A better understanding of the role of reliance in family
adaptation will facilitate the development of more effective
treatment approaches and lay the foundation for more
effective self-management in paediatric chronic pain.
Additional file 1: PRISMA-P 2015 Checklist. This checklist is approved for
use with systematic review protocol submissions. (DOCX 32 kb)
BMG: Brian McGuire; BPS: British Psychological Society; ISPP: International
Symposium on Pediatric Pain; JIA: Juvenile idiopathic arthritis; LC: Line Caes;
LH: Lisa Hynes; PICO: Participants, Interventions, Comparisons, Outcome(s);
PPC: The Paediatric Pain Conference; PRISMA: Preferred Reporting Items for
Systematic Reviews and Meta-Analyses; PROSPERO: Prospective Register of
Systematic Reviews; QOL: Quality of life; SPPAC: Society of Pediatric
Psychology Annual Conference; SS: Sophia Saetes
Child & Youth PhD Programme at the National University of Ireland, Galway
This project is not currently funded. No funders, sponsors, or institutions
have had roles in developing this protocol.
Availability of data and materials
SS carried out the initial background research and conceived of the study. SS
also drafted the manuscript. LC and LH helped in drafting the manuscript or
revising it critically for important intellectual content. BMG and LC made
substantial contributions to the conception and design of the project,
including revising the manuscript. All authors gave final approval of the
version to be published.
Ethics approval and consent to participate
Consent for publication
The authors declare that they have no competing interests.
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