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Research Portfolio Submitted in Part Fulfilment of the requirements for the
Degree of Doctorate in Clinical Psychology
Volume 1 of 2
Emily Louise Howe
University of Bath
Department of Clinical Psychology
September 2014
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Word counts
Critical literature review abstract……………………………………….…......120
Service improvement project abstract…………………………………….......238
Main research project abstract…………………………………………….…..237
Critical literature review………………………………………………….....…6877
Service improvement project…………………………….……………..….....5140
Main research project……………………………………………………....…6065
Executive summary……………………………………………………….....…845
Connecting narrative……………………………………………………....…2974
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Table of Contents
Acknowledgements…………………….…………………………………….……4
Abstracts……………………………………………………………………..……...6
Critical literature review……………………………………………………..…......9
Introduction…………………………………………………………..…….10
Method…………………………………………………………………..….13
Results………………………………………………………………...……24
Discussion…………………………………………………………………35
References…………………………………………………………...……41
Service improvement project…………………………………………………..…57
Introduction………………………………………………………………...58
Method……………………………………………………………………...61
Results………………………………………………………………...……65
Discussion…………………………………………………………………74
References……………………………………………………………...…79
Lay summary………………………………………………………………83
Main research project………………………………………………………..……86
Introduction…………………………………………………………...……87
Method……………………………………………………………...………92
Results……………………………………………………………...………100
Discussion…………………………………………………………...……108
References……………………………………………………………..…115
Executive summary………………………………………………………………128
Connecting narrative……………………………………………………….……131
Appendices………………………………………………………………….……140
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Acknowledgements
I would like to express great thanks to the research supervisors for my main
project; Dr Anna Lagerdahl and Professor Paul Salkovskis for their guidance,
encouragement and support throughout the project has been invaluable. Anna
in particular has allowed me to develop in my role as a researcher and clinician.
I would also like to thank Dr Jackie MacCallam and Dr Claire Lomax, researcher
supervisors of my service improvement project. They have been indispensable
in support of my development as a researcher and in the realisation of the
project. Particular thanks are extended to Jackie, whose tireless efforts in
recruitment of a hard-to-reach population have been invaluable. I would like to
express my thanks to Dr Jenny Rushforth, Clinical Psychologist at the Swindon
CAMHS service for commissioning and supporting my service consultancy
project. I am grateful to all of the clinicians for taking part in interviews and
offering their views and experiences of the eating disorder clinic. I would also
like to thank Dr Maria Loades for her supervision and enthusiasm for the project,
and Emelien Waite, Office Manager at Swindon Community CAMHS for her
unwavering support through difficult IT and technical difficulties.
This research would have not been possible without the participants whom I am
considerably grateful to for their interest, time, and valuable contributions to the
research. I would like to thank Great Western Hospitals Foundation Trust,
Salisbury NHS Foundation Trust and Sirona CIC for hosting my research. At
Great Western Hospital, I would like to thank Clinical Nurse Specialists Lynda
Lark and Jasmine Hebden. At Salisbury District Hospital, special thanks goes to
Clinical Nurse Specialists Shirley Holmes and Sonja Dabill, Consultant
Haematologist Dr Jonathon Cullis for his support and promotion of the project
within his team.
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I would also like to thank the Clinical Psychology Department administration
team at the University of Bath, particularly Jane French for receiving and
handling the many postal responses from participants.
I am ever grateful to colleagues in my cohort for their ongoing support and
interest in my research. I am thankful to the University of Bath for providing the
opportunity to undertake a broad range of clinically relevant research.
I would like to express my appreciation to Macmillan Cancer Voices for allowing
me to advertise my research as an opportunity for their members, and local
support groups through which I have gained a vast number of participants.
Finally, special thanks to my fiancé, Steve, who offered encouragement and
support alongside friends and family throughout my endeavours.
5
Abstract: Critical literature review
Despite a growing evidence base for the efficacy of treatments for comorbid
anxiety disorders in children with Autism Spectrum Disorders (ASD), empirically
grounded psychological models are limited and varied in nature. The current
review provides a synthesis and critical evaluation of the literature regarding
psychological models of anxiety in children and adolescents with ASD. A
systematic search of PsycINFO and Medline was performed yielding 771
articles; 18 of which met inclusion criteria. Studies were synthesised in two
categories; those derived from models of anxiety in typically developing children
and those related to core ASD symptoms. Research on theoretically robust
psychological models of anxiety in children with ASD is in its infancy.
Implications for clinical practice and future research are discussed.
Keywords: Autism spectrum disorders, ASD, anxiety, psychopathology,
comorbidity, models.
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Abstract: Service improvement project
Emotional distress is greater among parents of children newly diagnosed with a
life-limiting condition compared with the normal population. This study explored
parents’ experiences of emotional support following their child’s diagnosis of a
life-limiting condition and information received about common psychological
responses and emotional support. Five parents of children with a life-limiting or
life-threatening condition were recruited from a UK nursing and Psychology
complex health care service. Semi-structured interviews were conducted and
analysed using thematic analysis. Results revealed themes concerning
understanding what psychological support is available, what it involves and how
it can be accessed. A further theme concerned lack of information provision
about practical and emotional support with timeliness of such support as a
pertinent theme. Another theme related to ‘knowing what’s normal’ in terms of
emotional reactions to their child’s diagnosis and treatment. A theme of parents
prioritising the child’s needs over their own was also evident. This study
highlighted challenges that need to be addressed in order to improve provision
of information about psychological and emotional support for parents. Describing
the role of psychological and emotional support with a multi-disciplinary team,
how it can be accessed and what it might involve are key issues. Normalisation
of parents’ emotional responses throughout the child’s care, and consideration
of fathers’ needs are also important. Findings will be used to develop an
information leaflet for parents covering these issues, and to inform staff training
on responding to emotional needs.
Keywords: Parents, Life-limiting condition, Emotional support, Psychological
support, Qualitative
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Abstract: Main research project
Purpose: Research investigating psychological predictors of health anxiety and
related outcomes is lacking. This study investigated whether mental defeat,
existential concerns, beliefs about emotions and intolerance of uncertainty
predict levels of health anxiety, quality of life, depression and anxiety in cancer
patients in remission.
Method: A quantitative prospective design was employed. Ninety participants
aged 23-80, who had completed cancer treatment with curative intent were
recruited from two hospitals, support groups and the Macmillan website. Self-
report questionnaires were used to measure mental defeat, existential concerns,
beliefs about emotions and intolerance of uncertainty, health anxiety, quality of
life, depression and anxiety at two time points, 4 weeks apart.
Results: Clinically significant levels of health anxiety were reported in 52.2% of
the sample. Elevated health anxiety at Time 1 (T1) was significantly associated
with intolerance of uncertainty. Quality of life at T1 was significantly associated
with mental defeat and beliefs about emotions. Psychological distress at T1 was
significantly associated with mental defeat and intolerance of uncertainty.
Stepwise regressions demonstrated that mental defeat was a significant
predictor of health anxiety (including avoidance and reassurance seeking),
quality of life and distress 4 weeks later.
Conclusions: This study provides evidence that clinically elevated health anxiety
is high in cancer survivors, and highlights the importance of consideration of the
risk factors underlying elevated health anxiety, psychological distress and poor
quality of life that are appropriate targets for treatment. Clinical and research
implications are discussed.
Keywords: Cancer, curative, survivors, predictors, health anxiety, mental defeat
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Critical literature review
Empirically grounded psychological models of anxiety in Autism Spectrum Disorder: Mind the gap?
Emily Louise Howe
Doctoral Programme in Clinical Psychology, Department of Psychology,
University of Bath, Claverton Down, Bath, BA2 7AY, Tel: 01225 385506, Email:
Word count: 6507
May 2014
Internal/academic Supervisor: Dr Ailsa Russell, Department of Psychology, 6
West 0.2, University of Bath, Claverton Down, Bath, BA2 7AY,
Tel: 01225 385 517, Email: [email protected]
Proposed journal: Review Journal of Autism and Developmental Disorders.
This journal is aimed at an international audience and publishes critical reviews
across interdisciplinary research fields of autism spectrum disorders. It accepts
review topics across the lifespan analysing research trends which includes
comorbid conditions.
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Introduction
Anxiety disorders in children and adolescents with ASD
Anxiety disorders, or clinically elevated symptoms of anxiety are common in
children with Autism Spectrum Disorders (ASD) (de Bruin et al, 2007) with rates
of disorders ranging from 11 to 84% (White, Oswald, Ollendick & Scahill, 2009).
Recent changes to diagnostic criteria for ASD in the DSM-5 (American
Psychiatric Association, 2013) involved removal of individual categories such as
autistic disorder, pervasive developmental disorder not otherwise specified
(PDD-NOS), and Asperger syndrome; introduction of coding of disorder
severity; collapsing of communication and social behaviour into one domain;
changes in the number of symptoms required for diagnosis; and finally the
introduction of a new disorder, social (pragmatic) communication disorder. Prior
to these changes, researchers studied differing patterns of prevalence of anxiety
across diagnostic groups. Children with Asperger’s syndrome are more likely to
experience anxiety, followed by Pervasive Developmental Disorder Not
Otherwise Specified (PDD-NOS) and children with Autism (White, Oswald,
Ollendick & Scahill, 2009). Some authors have hypothesised that higher levels
of insight and self-awareness in individuals with Asperger’s syndrome and High
Functioning Autism (HFA) may account for these between-group differences
(e.g. Bellini, 2006a).
Studies of the nature of anxiety in this group have identified social anxiety,
obsessive compulsive disorder (OCD), specific phobia and generalised anxiety
disorders as particularly prevalent (Ollendick, King & Muris, 2004; van Steensel,
Bogels & Perrin, 2011). However, individuals with ASD may have difficulties in
describing feelings and emotions, so anxiety disorders may be masked by
autistic symptoms. It is therefore challenging to determine the most appropriate
diagnosis and treatment plan (Mazzone, Ruta & Reale, 2012). Comorbid anxiety
disorders are associated with significant distress and functional impairment
(Kelly, Garnett, Attwood & Peterson, 2008). Furthermore, higher levels of anxiety
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are hypothesised to exacerbate core ASD symptoms and behavioural problems
(Green, Gilchrist, Burton & Cox, 2000; Wood & Gadow, 2010; Farrugia &
Hudson, 2006), for example impacting on social responsiveness and social skills
in young people with ASD (Bellini, 2004).
Treatment efficacyResearch evaluating the applicability and efficacy of treatment for anxiety in this
population has been slow to emerge. However, particular promise has been
shown for Cognitive Behavioural Therapy (CBT) (Chalfant et al, 2006; Reaven &
Hepburn 2003; Sofronoff et al, 2005; Sze & Wood 2007; Wood et al, 2009). A
systematic review of nine studies of CBT for anxiety disorders concluded that
CBT was an effective treatment for anxiety in individuals with ASD (Lang,
Regester, Lauderdale, Ashbaugh & Harring, 2010). The studies reviewed used
CBT based on the models and protocols in existence for typically developing
children, including components such as psychoeducation, cognitive
restructuring, self-talk, relaxation, and exposure to feared stimuli. Research has
also focused on adaptations to standardised CBT for children and adolescents
with ASD. A review by Moree and Davis (2010) noted the following
recommendations; adjusting treatment to the child’s developmental level, use of
concrete and visual materials, relating work to the child’s specific special
interest(s), using a more directive approach and involving parents or carers.
Longer treatment duration is also recommended.
Theories of elevated prevalence of anxiety in ASDDespite the high prevalence of anxiety disorders in ASD, and an emerging
clinical framework for treatment, there is a paucity of empirically grounded
models of factors that underlie the development and maintenance of anxiety
disorders in ASD, and how they may interact with the core deficits of ASD.
Indeed, historically it was thought that anxiety may be part of ASD. However,
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more recent work has concluded that this is not the case (e.g. Kerns & Kendall,
2012). However, it is notable that in early work Kanner (1943) advocated that
many core features of ASD, particularly insistence on sameness and restricted
behaviours and interests were driven by anxiety. Kanner stated that ‘the child’s
behaviour is governed by an anxiously obsessive desire for the maintenance of
sameness’ (1943, p245). It has been hypothesised that the high comorbidity rate
of anxiety in ASD is likely to be explained by ASD deficits that make the
individual more prone to develop anxiety disorders. However, diagnostic overlap
may also partly explain elevated levels of anxiety (White, Oswald, Ollendick &
Scahill, 2009). It is unclear whether core processes and behaviours associated
with core features of ASD such as insistence on sameness and repetitive
behaviours are consequences of, or strategies to cope with anxiety (Gillott,
Furniss and Walter, 2001).
Research is now focusing broadly on two areas; determining which processes in
typically developing children and adolescents are present in those with ASD and
those that are unique to ASD, thought to be related to core symptoms (Ollendick
& White, 2012). Some studies also seek to explain the interaction of these
factors. The few accounts that do exist concern either neurobiological or
psychological theories of development. Psychological models are varied in their
focus, with the lack of consensus causing difficulties in translating them into
standardised, empirically grounded and testable treatments. Furthermore,
treatments of anxiety in this population are currently based on extant models of
anxiety in neurotypical children rather than derived from the phenomenology of
anxiety in ASD. For example, some researchers have focused on developmental
pathways of anxiety in ASD. Vasey and Dadds (2001) hypothesised that an
interaction of predisposing and protective factors account for the development,
maintenance and reduction of anxiety in individuals with ASD. Tantam (2000)
hypothesises that a high rate of adverse life events, victimization, awareness of
differences to peers, rumination, and relationship difficulties may account for the
high level of anxiety and depression in ASD.
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In line with the general consensus of psychopathology research as a field,
Rapport (2001), advises of the importance of theoretically derived treatment
targets and testable models. Therefore, it is of critical importance to examine
models of anxiety in children with ASD, in order to move towards an empirically
based understanding to inform accurate assessment, case conceptualisation,
and robust theoretically grounded treatments through future research. There are
currently no reviews of psychological models of anxiety in young people with
ASD. Previous reviews have tended to focus on prevalence of anxiety disorders
and other comorbidities, measures used to assess comorbidity or treatment
efficacy (e.g. Kim, Szatmari, Bryson, Streiner & Wilson, 2000; Matson & Nebel-
Schwalm, 2007; MacNeil, Lopes & Minnes, 2009).
The current review therefore aims to address this need by providing a critical
overview and synthesis of the published literature in this area, leading to
identification of areas for future research. It aims to contribute to stimulating the
development of comprehensive, empirically sound psychological models on
which treatment guidelines and protocols can be based.
MethodLiterature searchThe current review adhered to guidelines outlined in the Preferred Reporting
Items for Systematic reviews and Meta-Analyses (PRISMA) statement (Moher,
Liberati, Tetzlaff & Altman, 2009). An extensive systematic literature search was
conducted in order to locate published articles concerning psychological models
of anxiety in children with ASD. Key word searches of articles indexed in the
PsycINFO and Medline databases were performed on 29th November 2013. The
date range of the search was limited to articles published between 1980 and 29 th
November 2013; in line with the development of the field since the inclusion of
clear diagnostic criteria for Autism in the DSM-III in 1980 (American Psychiatric
13
Association, 1980). The following search term combinations were used
concurrently to search both databases; autism OR Asperger* OR "pervasive
developmental disorder" OR "social communication disorder" AND anxiety OR
"Social phobia" OR "Obsessive compulsive disorder" OR OCD OR panic OR
"psychiatric disorder" OR psycholog* AND model OR theory OR pathway OR
development OR maintenance OR factor OR predictor OR cause OR aetiology
OR framework OR account OR association OR mechanism OR relationship OR
association.
Inclusion/exclusion criteriaEach article was screened for inclusion according to the following criteria; the
article pertained to psychological models or correlates of anxiety in children
(aged 0-18), the article was classified as primary research with a clinical
population, the article was published in a peer-reviewed journal, the article was
published in English language and was published from 1980 onwards. Book
chapters, conference abstracts, case studies, review articles, epidemiological
studies and dissertation and theses abstracts were excluded.
Selection of studiesThe initial search identified 773 articles. Duplicates were removed by the first
author, resulting in a remaining 771 articles. The abstracts were reviewed by
the first author for inclusion. Eighteen studies met inclusion criteria. One study
(Boulter et al, 2014) was published as an early view article online, and was
therefore within the date range at the time of review, and published fully in 2014.
The flow of articles through phases of the literature review is summarised in
Figure 1.
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Figure 1.Flow of articles through consecutive phases of the literature review.
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Table 1.Characteristics of the studies included in the systematic review.
Author(s) (year) Aims Sample MethodBellini (2004) To examine
prevalence and types of anxiety in adolescents with HFAand factors relatedto anxiety, specifically social skills deficits.
N= 41 adolescents;Asperger’s syndrome: n=16PDD-NOS: n=6(11 diagnosed with anxiety, 6 taking medication for anxiety). Mean age: 14.22 years.
Cross sectional questionnaire design.Adolescents completed the Social Skills Rating System (SSRS; Gresham & Elliot, 1990), the Multidimensional AnxietyScale for Children (MASC; March,1999), and the Social AnxietyScale for Adolescents (SAS-A; La Greca,1999)Parents completed the SSRS and the Behavior Assessment System for Children (BASC; Reynolds & Kamphaus, 1992).
Burnette, Mundy, Meyer, Sutton, Vaighan & Charak (2005)
To investigate relations between the weak central coherence hypothesis, theory of mind skills, andsocial-emotional functioning in children with ASD.
N = 26 children with HFA and N=33 matched controls.Mean age: 11.25 years.
Experimental design.Participants completed the WechslerIntelligence Scale for Children—Third Edition(WISC-III; Wechsler, 1991), a first and second order Theory of Mind (ToM) task (M&Ms false belief task; Ozonoff, Pennington, & Rogers, 1991a; second-order ToM task (Baron-Cohen,1989; Ozonoff, Pennington, & Rogers, 1991a) to assess social-cognitive abilities and a battery of tests to assess Weak Central Coherence (Block Design Subscale of the Wechsler Intelligence Scale for Children—Third Edition (WISC-III; Wechsler, 1991), the Differential Abilities Scale—Pattern Construction subtest (DAS; Elliott, 1990), the Embedded Figures Test (EFT; Benton & Spreen, 1969), and the modified homograph task (e.g. Happe, 1997; Joliffe &Baron-Cohen, 1999). Social-emotional functioning was measured using The
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Behavioral Assessment System for Children Self-Report of Personality (BASC-SRP; Reynolds & Kamphaus, 1998) and the Social Anxiety Scale for Children-Revised (SASC-R: La Greca & Stone, 1993).
Bellini (2006) To examine thecontribution of social skill deficits and physiological hyperarousal to the development of social anxiety in adolescents with ASD.
N= 41 adolescents with ASD;Autism: n=19Asperger syndrome: n=16PDD-NOS: n=6Mean age: 14.22 years.
Cross sectional questionnaire design.Participants completed The Social Skills Rating System (SSRS; Gresham & Elliot, 1990), The Social Anxiety Scale for Adolescents (SAS-A; La Greca,1999), and The Multidimensional Anxiety Scale for Children (MASC; March 1999).
Ben Sasson et al (2008)
To determine patterns of sensory modulation dimensions of sensory clusters of toddlers with ASD. To investigate whether there is a sensory-based subgroup that has higher levels of affective symptoms.
N=170 toddlers with ASD.Mean age: 28 months.
Cross sectional cluster analysis design.Parents completed the Infant Toddler Sensory Profile (Dunn,2002) under-responsivity, over-responsivity, and seeking scales and Infant Toddler Social Emotional Assessment (Carter & Briggs-Gowan, 2005).
Sukhodolsky et al (2008)
To examine frequency and correlates of parent-rated anxiety symptoms in children with Pervasive Developmental Disorder (PDD).
N= 171 children with PDD.Mean age: 8.2 years
Cross sectional questionnaire design.Parents completed twenty items of the Child and Adolescent Symptom Inventory (CASI; Gadow & Sprafkin 1994, 2002; Gadow & Sprafkin 1997, 1998).
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Davis et al (2011)
To investigate whether or not communication deficits differentiallyaffect children with ASD compared to those without ASD.
N= 99 children;Autistic disorder: n=33PDD-NOS: n=33No diagnosis: n=33Mean age: 7.5 years.
Cross sectional questionnaire designA composite symptom checklist from the Diagnostic and Statistical Manual – Fourth Edition – Text Revision (DSM-IV-TR; APA, 2000) and the International Classification of Diseases, Tenth Edition (ICD-10; World Health Organization, 1992) was used to define criteria for the various ASDs.The Autism Spectrum Disorders- Diagnostic for Children (ASD-DC; Matson and Boisjoli 2009) was used to measure verbal communication.The worry/depressed and avoidant behaviour subscales of The Autism Spectrum Disorders- Comorbidity for Children (ASD-CC; Matson & Wilkins, 2008) were combined for the purposes of this study to provide a measure of anxiety.
Davis et al (2012)
To determine whether deficitsin communication skills have an effect on the expression ofanxiety in infants and toddlers with autistic disorder andPDD-NOS.
N=735;Autistic Disorder (n=107),PDD-NOS: n=110 Atypically developing children with no diagnosis of Autistic Disorder (AD) or PDD-NOS: n=518
Mean age: 26.1 months.
Cross sectional questionnaire design.Using clinical judgment, diagnoses were made based on the Diagnostic and Statistical Manual of Mental Disorders, Fourth Edition, Text Revision (DSM-IV-TR; American Psychiatric Association, 2000) criteria for AD. The Modified Checklist for Autism in Toddlers (M-CHAT; Robins, Fein, Barton, & Green, 2001) and Battelle Developmental Inventory-Second Edition (BDI-2; Newborg, 2005) were used to inform diagnosis for PDD-NOS.The avoidance behaviour and anxiety/repetitive behaviours subscales from Part 2 of the Baby and Infant Scale for Children With aUtIsmTraits (BISCUIT; Matson et al, 2009) were combined to provide a total anxiety score.The communication domain and receptive and expressive communication subdomains from the Battelle Developmental Inventory-Second Edition (BDI-2; Newborg, 2005) were used to assess different forms of communication.
Rieske, Matson, May & Kozlowski (2012)
To investigate differences in anxiety symptoms betweenHFA and Asperger’s syndrome in
N= 181 children;Autistic Disorder: n=59 Asperger’s
Cross sectional questionnaire design.The Autism Spectrum Disorders- Diagnostic for Children (ASD-DC; Matson and Boisjoli 2009) and The Autism Spectrum Disorders- Comorbidity for Children (ASD-CC; Matson & Wilkins, 2008) were used to diagnose ASDs.A total anxiety score was created by combining the Worry/Depressed and
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comparisonto typically developing controls, specifically examining the possible moderating effect of social deficits.
Syndrome:n=49Typically Developing children: n=73
Mean age: 8.6 years
Avoidant Behavior subscales ofthe ASD-CC (Matson & Wilkins, 2008).A total social deficits score was created by combining the Nonverbal Communication/ Socialization and Social Relationships domains of the ASD-DC (Matson and Boisjoli 2009).
Spiker, Lin, Van Dyke & Wood (2012)
To explore the association between various modes of expression of Restricted Interests and anxiety disorder symptoms.
N= 68 children diagnosed with HFA(Autism, Asperger syndrome, or PDD-NOS)
Mean age: 9.4 years
Cross-sectional questionnaire design.The Yale Special Interest Survey (YSIS; Klin et al.,2007) was used to measure restricted interests.The Children’s Yale-Brown Obsessive Compulsive Scale (CY-BOCS Goodman et al, 1989) was used to measure obsessions and compulsions related to OCD.The Anxiety Disorders Interview Schedule (ADIS; Silverman and Albano, 1996) was used to measure symptoms of anxiety disorders.The Multidimensional Anxiety Scale for Children–Parent Version (MASC-P; March, 1997) was used to measure general anxiety.
Nidditch, Varela, Kamps & Hill (2012)
To investigate relationships between anxiety, aggression, social understanding, IQ, and diagnosis in children with ASD.
N=231 children with ASD;Autistic Disorder: n=140Asperger’s Disorder: n=81PDD-NOS: n=10
Cross sectional questionnaire design.The Autism Diagnostic Interview–Revised (ADI-R; Lord, Rutter, &LeCouteur, 1994) was used for diagnosis. The AutismDiagnostic Observation Schedule (ADOS; Lord, Rutter,DiLavore, & Risi, 2002) were used to inform ASD diagnosis.The Mullen Scales of Early Learning (Mullen, 1995), Wechsler Preschool and Primary Scale of Intelligence–Third Edition (Wechsler, 2002), Wechsler Intelligence Scale for Children–Fourth Edition (Wechsler, 2003), Leiter International Performance Scale–Revised (Roid & Miller, 1997) or Wechsler Nonverbal Measure of Ability (Wechsler & Naglieri, 2006) were used to assess intellectual ability, dependent on participants age and verbal ability. The Anxiety, Aggression, and Social Skills Subscales of the Parent Rating Scales (PRS) of the Behavioral Assessment System for Children–Second Edition (BASC-2; Reynolds & Kamphaus, 2004) were used to measure anxiety, aggression, and social understanding.
19
Rodgers, Riby, Janes, Connolly & McConachie (2011)
To compare anxiety in children with ASD and Williams Syndrome, specifically to examine the relationship betweenrepetitive behaviours and anxiety.
N= 54 children;ASD (n=34), mean age= 12.2 years Williams’ syndrome (n=20), mean age: 9.4 years.
Cross sectional questionnaire design.The Repetitive Behaviours Questionnaire (RBQ; Turner 1995,1999) was used to measure repetitive behaviours. The Spence Children’s Anxiety Scale—Parent (SCAS-P; Spence 1998) was used to assess anxiety.
Rodgers, Glod, Connolly & McConachie (2012)
To compare repetitive behavioursand anxiety in children with ASD with high and low anxiety, respectively.
N= 67 children with ASD.Mean age: 11.2 years.
Cross sectional questionnaire design.IQ was measured using the Wechsler AbbreviatedScales of Intelligence (WASI: Wechsler 1999).The Spence Children’s Anxiety Scale-Parent Version (SCAS-P; Spence 1998) was used to measure anxiety. Children were divided into two groups based on their anxiety score. The Repetitive Behaviours Questionnaire (RBQ; Turner, 1995) was used to assess Restricted and Repetitive behaviours.
Rieske, Matson, Davis & Thompson (2013)
To investigate relationships between cognitive and adaptive functioning and anxiety symptoms in infants and toddlers with ASD, particularly the moderating effect of autism symptomatology.
N=2336 infants and toddlers with ASD.Mean age: 25.7 months.
Cross sectional questionnaire design.ASD diagnoses had previously been confirmedbased on scores obtained on the Modified Checklist for Autism in Toddlers (Kleinman et al. 2008; Robins et al. 2001), the DSM-IV-TR criteria (APA 2000), the developmental profiles of the Battelle Developmental Inventory-2 (BDI-2; Newborg 2005), and clinical judgment.The adaptive and cognitive domain scores of the Battelle Developmental Inventory-Second Edition (BDI-2; Newborg 2005) were used to assess adaptive and cognitive functioning.The Baby and Infant Screen for Children with aUtIsm Traits-Part 1 (BISCUIT-Part 1; Matson et al. 2007a) was used to measure autism symptomology.The avoidance behaviour and anxiety/repetitive behaviour subscales of The Baby and Infant Screen for Children with aUtIsm Traits-Part 2 (BISCUIT-Part 2; Matson et al. 2007a, b) were combined to provide a measure of anxiety symptoms.
Gotham et al (2013)
To explore the association between
N=1429 children with
Cross sectional questionnaire design.The Autism Diagnostic Interview–Revised (ADI-R; Lord, Rutter, &
20
anxiety and ASD symptoms,particularly the degree to which the relationship is explained by insistence on sameness (IS) behaviours and/or cognitive ability.
ASD.Mean age: 1 year 2 months
LeCouteur, 1994) was used for diagnosis. The Autism Diagnostic Observation Schedule (ADOS; Lord et al., 2000),The Vineland Adaptive Behavior Scales, Second Edition (Vineland-II; Sparrow, Cicchetti, & Balla, 2005) andThe Social Responsiveness Scale (SRS; Constantino& Gruber, 2005) were used to support diagnosis.The Child Behaviour Checklist 4/18 (CBCL; Achenbach, 1991) was used to measure the child’s current social competence and emotional and behavioural problems, including anxiety.Exploratory factor analysis of the ADI-R Stereotyped and Repetitive Behavior items yielded an ‘insistence on sameness’ factor comprised of Difficulty with Minor Changes in Routine, Compulsions/Rituals, Resistance to Trivial Changes in the Environment, Abnormal Response to Specific Sensory Stimuli, Sensitivity to Noise, and Circumscribed Interests. This factor was used as a measure of insistence on sameness.
Stratis & Lecavalier (2013)
To investigatethe relationship between restricted and repetitive behaviours and co-occurring psychiatric symptoms, including depressive, anxiety, attention-deficit hyperactivity disorder (ADHD) andoppositional defiantdisorder (ODD) symptoms, specifically considering the role oflevel of functioning.
N=72 children with ASD.Mean age: 11 years.
Cross sectional questionnaire design.The Social Communication Questionnaire (SCQ; Rutter, Bailey, & Lord, 2003) was used to measure ASD symptoms.The Repetitive Behavior Scale-Revised (RBS-R; Bodfish, Symons, & Lewis, 1998) was used to measure frequency and severity of restricted and repetitive behaviours across the 5 domains (Ritualistic/Sameness Behavior, Self-Injurious Behaviour, Stereotypic Behavior, Compulsive, Behavior and Restricted Interests).The Child Symptom Inventory-4 (CSI-4; Gadow & Sprafkin, 2002) was used to assess anxiety disorders.The Adaptive Behavior Assessment System, 2nd Edition (ABAS-II; Harrison & Oakland, 2003) was used to assess level of functioning.
21
Hollocks, Ozsivadjian, Matthews, Howlin & Simonoff (2013)
To investigate whether an attentional bias towards threatening information is present in young people with ASD and anxiety symptoms.
N= 49 children aged 10-16 years;ASD (n=38)Typically developing controls (n=41).
Experimental design.Two versions of the dot-probe paradigm, the first with emotional faces and the second with emotional word were used to measure attentional bias. The faces were taken from the NimStim face set (Tottenham et al, 2009). Autism-specific anxiety words were derivedfrom a qualitative study (Ozsivadjian, Knott, & Magiati, 2012), which investigated overlaps as well as differences in the presentation of anxiety in ASD and the general population.The Spence Children’s Anxiety Scale (parent and child versions, SCAS-P and SCAS-C, Spence, 1998) was used to measure anxiety symptoms.The Children’s Depression Inventory (CDI; Kovacs, 1992) was used to assess symptoms of depression.The Social Communication Questionnaire (SCQ; Rutter, Bailey, & Lord, 2003) SCQ was used to assess social and communication skills.The two subtest version of the WASI (Wechsler, 1999) was used to measure intelligence.The word reading test from the WIAT (Wechsler, 2005) was selected as a brief measure of reading ability.The affect recognition and inhibition subtests of the NEPSY-II (Korkman, Kirk, & Kemp, 2007) were used to measure participants’ ability to recognize emotions in faces (happy, sad, anger, fear, disgust and neutral) and to measure ability to inhibit automatic responses to stimuli.
Eussen, Van Gool, Verheij, De Nijs, Verheulst & Greaves-Lord (2013)
To examine associations between quality ofsocial relations, symptomseverity, intelligence and anxiety in children with ASD.
N=134 children with ASD (58 with a comorbid anxiety disorder).Mean age: 9.2 years.
Cross sectional questionnaire design.The Child Behaviour Checklist 4/18 (CBCL; Achenbach, 1991) was used as a continuous measure of anxiety.The DSM-oriented scale ‘Anxiety Problems’ (Achenbach and Rescorla, 2001) was used as a measure of anxiety.The ‘social relationships’ subscale of the Children’s Communication Checklist (CCC; Bishop, 1998) was used to obtain an overall measure of quality and number of social relations.The ADOS module 3 (Lord et al, 1999) was used to assess ASD symptom severity.The Dutch version of the WISC-R was administered (Wechsler,
22
1974; WISC-R Project Group, 1986) as a measure of intelligence.Boulter, Freeston, South & Rodgers (2014)(NB: Early view publication online within date criteria)
To replicate the relationship between intolerance of uncertainty and anxiety in typically developing children and adolescents and establish whether this relationship is present in children with ASD.
To compare the relationship between intolerance of uncertainty and anxiety in the ASD and typically developing groups.
N=224 children and adolescents;ASD (n=114), typically developing controls (n=110).Mean age: 12.8 years.
Cross sectional questionnaire designThe Social Responsiveness Scale (SRS; Constantino, 2002) was used to confirm ASD diagnosis in the ASD group.The Intolerance of Uncertainty Scale: Child and ParentVersions; (IUS-C; Walker 2009; IUS-P; Rodgers et al,2012) was used to measure levels of intolerance of uncertainty.The Spence Children’s Anxiety Scale Child and ParentVersions (SCAS-C; Spence 1998; SCAS-P, Nauta et al.2004) was used to measure anxiety symptoms.
23
ResultsThis section seeks to synthesise key findings of the studies included in the
current review in order to establish whether there are theoretically grounded and
complete models of anxiety in children and adolescents with ASD. Research has
broadly focused on two areas; correlates of anxiety observed in typically
developing children and adolescents that may be applicable to those with ASD,
and those related to core symptoms of ASD. Some studies have focused on the
interaction of the two. Articles have therefore been categorised into these three
areas, and subdivided into themes based on the focus of the study.
Correlates of anxiety in typically developing children
Attentional bias
An attentional bias towards threatening information has been well established as
a cognitive process in anxiety in typically developing children and adolescents
(e.g. Lau et al, 2012). Hollocks, Ozsivadjian, Matthews, Howlin and Simonoff
(2013) examined whether this bias was present in children and adolescents with
anxiety symptoms and ASD. The dot-probe paradigm was used, firstly with
emotional faces and secondly with emotional words. The sample comprised 38
males with ASD and 41 neurotypical controls aged 10-16 years. Participants
with ASD had significantly higher levels of self-reported and parent-rated anxiety
and depression compared with controls. However, there was no significant
relationship between anxiety and attentional bias in either task, for either group
even when participants were divided into high and low anxiety. Interestingly, this
study demonstrated that high levels of anxiety in young people with ASD may
not be associated with attentional bias to threat. This suggests that attential bias
to threat, a common process implicated in the typically developing population
may not be present in young people with ASD. However, methodological
limitations are present. Hollocks, Ozsivadjian, Matthews, Howlin and Simonoff
(2013) highlight that it is interpretational biases, as opposed to attentional biases
are more associated with anxiety in ASD, which are also present in models of
24
anxiety in typically developing children. Furthermore, the standardised dot-probe
paradigm may not be targeting the idiosyncratic anxieties of children with ASD,
which threatens the validity of the study.
Mixed results in the literature have given rise to a debate concerning whether
the phenomenology of attentional biases are comprised of facilitated attention
and/or a difficulty in disengagement from threat information (Cisler & Koster,
2010). Hollocks, Ozsivadjian, Matthews, Howlin and Simonoff (2013) used bias
scores to assess attentional biases, which are calculated by deducting the mean
reaction time (RT) for congruent trials from the mean RT for incongruent trials.
This approach lacks a baseline on which the data is centred, meaning it is not
possible to determine what speed of RTs mean in relation to baseline attention.
This limits the conclusions that can be drawn from this study.
Intolerance of uncertainty
Intolerance of uncertainty (IOU) is a psychological construct that has been
implicated in anxiety disorders in typically developing child and adult populations
(see Gentes & Ruscio, 2011 for a review). IOU is defined as a cognitive bias that
affects how a person perceives, interprets, and responds to uncertain situations
(Freeston et al, 1994). Boulter, Freeston, South and Rodgers (2014)
investigated the role of IOU in children and adolescents with Autism or
Asperger’s syndrome. At the time of this review, this was the only study
investigating the construct of IOU in relation to ASD. It was an early view
publication published online, and was therefore within the date range specified.
The authors specifically sought to compare the association between IOU and
anxiety in typically developing children and adolescents and those with ASD.
Results indicated that IOU was significantly related to anxiety in both typically
developing children and those with ASD. IOU was found to mediate the
relationship between ASD and anxiety, and there was no difference between
diagnostic groups. The authors proposed a causal model in which ASD is
associated with greater intolerance of uncertainty, which leads to higher levels of
25
anxiety and repetitive behaviours. Core features of ASD such as rigidity of
thought difficulties with emotion processing are also thought to lead to elevated
IOU. These findings therefore support the hypothesis that elevated anxiety in
children with ASD may be explained by similar processes as observed in
typically developing children. This study has methodological strengths in terms
of the large sample size and specific measures used. It is also a strength of the
study that it proposes a testable causal model. However, as noted by the
authors, the degree of certainty perceived by individuals with ASD may be
impacted by core ASD symptoms such as social communication difficulties and
perceived uncertainty in social situations. This study offers unique insight into
the mediating role of IOU in the expression of anxiety in ASD. The
generalisability of the results may be limited however by the fact that the sample
comprised participants with HFA, without consideration of other diagnostic
groups which may have differing levels of awareness of perceived uncertainty
and IOU. The concept of IOU has resonance with insistence on sameness and
repetitive behaviours, discussed in the next section.
Core symptoms of ASD
Restricted interests / repetitive behaviours
The association between restricted interests, repetitive behaviours and anxiety
in children and adolescents with ASD has received considerable attention in the
literature compared to other core ASD symptoms. A preoccupation with
restricted interests (RI) is a core symptom of autism spectrum disorders (ASD),
expressed through a variety of ways such as play, or fact collection. Obsessive
Compulsive Disorder (OCD) shows an overlap in symptom presentation with RI,
and prevalence is high in children with ASD (Simonoff et al, 2008). Spiker, Lin,
Enjey, Van Dyke and Wood (2012) explored the association between modes of
RI expression and anxiety disorder symptoms in children with HFA. Findings
indicated that symbolic enactment of RI in the form of play, rather than
26
information collection or time engaged in RI, was significantly associated with
the increased presence and severity of anxiety symptoms. Spiker, Lin, Enjey,
Van Dyke and Wood (2012) hypothesise that manifestations of RI that are more
intense or take on specific (symbolically enacted) forms could be used as a
coping strategy for anxiety. This study provides evidence that a core feature of
ASD (RI), specifically symbolic enactment, is associated with anxiety in young
people with ASD, adding to the argument that anxiety in this population is
related to factors not present in typically developing children and adolescents
with anxiety disorders. However, this study suggests that elevated anxiety is not
caused by RI, but RI is a result of increased anxiety, possibly as a coping
strategy. This is consistent with Boulter, Freeston, South and Rodgers’ (2014)
tentative model that suggests restricted and repetitive behaviours may represent
a coping strategy for dealing with heightened anxiety. However, Spiker, Lin,
Enjey, Van Dyke and Woods’ (2012) hypothesis is speculative, and it does not
explore how increased RI as a coping strategy may impact on the maintenance
of anxiety. Therefore, although this study reports an interesting finding relevant
to understanding the phenomenology of how children with ASD may cope with
anxiety, it does not contribute specifically to understanding of the causal factors
in the development and maintenance of anxiety in this population.
Similar phenomenology in the form of repetitive behaviours (RB) has been
implicated in anxiety in young people with ASD. Sukhodolsky et al (2008)
investigated a number of correlates of anxiety in children with PDD, including
stereotyped behaviours. The authors were particularly interested in the role of
level of functioning. Higher levels of anxiety were associated with higher IQ,
functional language use and higher levels of stereotyped behaviours. This
finding provides some support for these variables as correlates of anxiety, and
evidence of higher levels of stereotyped behaviours may be explained by use of
these as a coping strategy, as suggested by studies discussed thus far in this
section. However, this was not tested in this study. A limitation of this study
noted by the authors is that the limited sample size precluded the examination of
27
differences between diagnostic groups of autism, Asperger’s or PDD-NOS. It
has been observed that individuals with Asperger syndrome, HFA and PDD-
NOS are more likely to experience anxiety (White, Oswald, Ollendick & Scahill,
2009). Furthermore, this study is correlational, meaning causation cannot be
inferred.
Rodgers, Riby, Janes, Connolly and McConachie (2011) conducted a cross
sectional comparison of anxiety and RB in children with ASD and William’s
syndrome. The sample comprised thirty-four children with autism aged between
8 and 16 years and twenty with Williams Syndrome aged between 6 and 15
years. There was a significant relationship between RB and anxiety in the ASD
group. This relationship was not found in the Williams’ syndrome group. Another
study by Rodger and colleagues (Rodgers, Glod, Connolly and McConachie,
2012) further explored the relationship between RB and anxiety in children with
ASD by comparing those with high anxiety and those with lower levels of
anxiety. The same sample and procedure was used as reported in the previous
study. Children were divided into the high and low anxiety groups according to
indicative cut-off scores on the SCAS-P (Spence, 1998). Children within the high
anxiety group had higher levels of RB, insistence on sameness (IS) and
circumscribed interests and sensory-motor behaviours than those with lower
levels of anxiety. Furthermore, within the high anxiety group, higher levels of
insistence on sameness/circumscribed interests, but not sensory-motor
behaviours were associated with higher anxiety. This relationship was not
observed in the low anxiety group. The results of this study support the findings
of Spiker, Lin, Enjey, Van Dyke and Wood (2012), providing further support for
the relationship between RB and anxiety, specifically the role of IS, which is
similar to IOU. Again, the high anxiety group may have developed stronger IS
and/or circumscribed interests to cope with anxiety, as speculated by Spiker, Lin,
Enjey, Van Dyke and Wood (2012).
28
Insistence on sameness
Gotham et al (2013) further contributed to this line of research, exploring the
association between anxiety and ASD symptoms in terms of the degree to which
IS behaviours account for anxiety. The sample included 1429 individuals
diagnosed with ASD aged 5 - 18 years. Exploratory factor analysis confirmed an
IS factor comprised of Difficulty with Minor Changes in Routine,
Compulsions/Rituals, Resistance to Trivial Changes in the Environment,
Abnormal Response to Specific Sensory Stimuli, Sensitivity to Noise, and
Circumscribed Interests. There was a minimal, yet significant association
between IS and anxiety. Neither anxiety nor IS were associated with other core
autism diagnostic scores. This demonstrated that anxiety and IS appear to
function as distinct constructs, each with a wide range of expression in children
with ASD across age and IQ levels. This indicates that there is a weak
relationship between IS and anxiety in ASD. Gotham et al’s (2013) findings
therefore do not fully concur with previous research described in this section.
The difference in choice of measures of IS may provide some explanation for
the differing pattern of results, but the use of factor analysis is a strength of this
study. Furthermore, Rodgers, Glod, Connolly and McConachie (2012) only
observed the relationship between IS and anxiety in those with higher levels of
anxiety, and Gotham et al (2013) did not differentiate levels of anxiety, which
may account for why a stronger relationship was not observed.
Stratis, Lecavalier and Luc (2013) investigated the relationship between
restricted and repetitive behaviours and co-morbid psychiatric symptoms,
including anxiety, in children with ASD. Similarly to Sukhodolsky et al’s (2008)
study, the role of level of functioning was examined. Parents of children aged 5-
17 diagnosed with ASD completed measures of social communication, repetitive
behaviour, anxiety symptoms and adaptive behaviour. Findings indicated that
ritualistic and sameness behaviour predicted higher levels of anxiety, in contrast
to stereotypy, which predicted Attention Deficit and Hyperactivity Disorder
29
(ADHD). Interestingly, level of functioning did not moderate the relationship
between ritualistic and sameness behaviour and anxiety, but was a moderator
for ritualistic and sameness behaviour and depressive symptoms. In line with
research already discussed in this section, this study provides further support for
the hypothesis that anxiety in ASD is associated with insistence on sameness
specifically, as opposed to restricted or repetitive behaviours per se.
ASD symptoms
Rieske, Matson, Davis and Thompson (2013) conducted a large scale study
examining whether autism symptomology in general moderated the relationship
between ASD and anxiety in infants and toddlers. Autism symptomology was
found to be correlated with anxiety, as was cognitive and adaptive development.
Autism symptomatology also moderated the relationship between cognitive and
adaptive development with anxiety but with a small effect size. This study points
to a positive relationship between autism symptomatology and anxiety in infants
and toddlers. However, it is difficult to draw comparisons with other studies
included in this review, as it has a broad level of analysis, focusing on autism
symptomology which may have a wide range of expression, and is the only
study concerned with infants. Further research is therefore warranted in this
area, to delineate the mechanisms at play in the relationship between autism
symptomology and anxiety in young children with ASD. Nonetheless, it does
strengthen the hypothesis that factors unique to ASD play a part in elevated
anxiety, and that difficulties may develop at an early age. More longitudinal
research is needed to examine the development and trajectory of anxiety in ASD
over time.
Aggression, social understanding, intelligence, and diagnosis
Niditch, Varela, Kamps, Hill and Trenesha (2012) examined relationships
between anxiety and aggression, social understanding, intelligence and
diagnosis children with ASD aged 2-9. Higher social understanding and higher
30
levels of aggression were found to mediate the relationship between anxiety and
IQ in toddlers. Interactions between intelligence, social understanding and
aggression predicted anxiety in pre-school and elementary school aged children.
Authors concluded that the trend appears to be that the association between
intelligence and anxiety is increasingly driven by either aggression or social
understanding over the course of childhood. This indicates that higher levels of
social understanding (likely to be present in individuals with HFA) and
aggression, mediates the relationship between anxiety and intelligence, in that
those with higher social understanding are likely to feel anxious about their
aggression. This offers an important insight into factors implicated in anxiety in
ASD at a young age. The study relied on parent-report data, which is
understandable given the young age of participants, but multiple informants
would have strengthened the validity of this study.
Social communication and interaction
Social deficits
Social deficits, a central part of ASD, have been implicated in a range of ways in
anxiety and ASD in young people. Bellini (2004) investigated the prevalence,
types and correlates of anxiety in adolescents with HFA. More specifically, the
study sought to determine whether social skill deficits were associated with
social anxiety in this population. A cross-sectional questionnaire design was
used. Adolescents completed self-report measures of social skills, and parents
also completed measures of social skills and anxiety.
A low negative correlation was observed between social assertion skills and
social anxiety in that as social assertion skills decreased, social anxiety
increased. There was also a moderate relationship between empathic skills and
social anxiety. Bellini (2004) hypothesised that this relationship is likely to be bi-
directional, in that those with poor assertion skills may be more likely to
experience social anxiety, and those with high social anxiety may be less likely
to initiate social interactions and therefore limit their ability to develop assertion
31
skills. However, these hypotheses were not tested in the study. The small
sample size of this study and no control group mean that the findings must be
interpreted with caution. Furthermore, the use of self-report measures to assess
anxiety and social skills is questionable given the difficulties inherent in ASD of
recognising and labelling emotions and reflecting on social situations. However,
this study provides specific support for the role of social skills deficits in social
anxiety in adolescents with ASD, which needs further exploration.
Rieske, Matson, May and Kozlowski (2012) compared anxiety symptoms in
children and adolescents with HFA, Asperger’s syndrome and a typically
developing control group. The study explored the possible moderating effect of
social deficits within these groups. The HFA and Asperger’s groups did not differ
in levels of anxiety compared with controls. Contrary to predictions, social
deficits did not significantly moderate the relationship between diagnosis and
level of anxiety. However, social deficits were found to moderate the relationship
with anxiety in those with Asperger’s syndrome but only approached significance
in those with HFA or Autism. This study had mixed results, providing support for
the moderating role of social skills deficits in anxiety in those with Asperger’s
syndrome, but not other groups. However, the validity of Asperger’s syndrome is
questionable, given the similarities with HFA. This is reflected in the recent
changes in criteria in the DSM-5 (American Psychiatric Association, 2013),
where Asperger’s is no longer included as a diagnostic category. Nonetheless,
this study gives support to the moderating role of social skills deficits in social
anxiety in young people with ASD.
Bellini (2006a) replicated and extended his previous study to propose a
developmental model of social anxiety in adolescents with HFA, examining the
contribution of social skill deficits and physiological hyperarousal. The model
proposes that temperament (including tendency for high physiological arousal)
leads to social withdrawal, which hinders development of social skills, thereby
precipitating negative interactions with peers, which triggers social anxiety.
32
Individuals with ASD will be adversely conditioned to negative social
interactions, due to high physiological arousal. This study employed a cross-
sectional questionnaire design, using parent and self-report measures of anxiety
and social skills. Multiple regression analyses were conducted, with anxiety as
an outcome variable and social skills and physiological arousal as predictors.
The proposed model was found to be a significant predictor of social anxiety.
This study provides evidence for the role of social skills deficits in the
development of social anxiety in adolescents with ASD.
Eussen et al (2013) explored the association between anxiety and quality of
social relations, symptom severity and intelligence in children with ASD. Results
indicated that elevated anxiety was associated with lower quality of social
relations and lower symptom severity. There was no relationship between levels
of anxiety and intelligence. However, the authors note that the sample was
comprised of only high-functioning children. No moderation effects were found.
In line with their findings that lower quality of social relations and lower symptom
severity were associated with elevated anxiety, Eussen et al (2013) point to the
possible value of developing therapeutic interventions for anxiety that improve
social relations, and that this may be of benefit to children with lower levels of
symptom severity. It must be noted that this study has limited generalisability in
terms of the sample used, which comprised those with normal levels of
intelligence and sufficient verbal ability to interact with others. Nonetheless, it
provides support for the hypothesis that features unique to ASD, in terms of
social deficits are likely to be at play in the development of anxiety in children
with ASD.
Communication deficits
Communication deficits have been shown to be associated with increased
anxiety in typically developing children (e.g. Blood et al, 2007). Two studies have
investigated the role of communication effects in anxiety in children with ASD
33
(Davis et al, 2011; Davis et al, 2012). Davis et al (2011) sought to examine
whether there were fundamental differences in the effects of communication
deficits on anxiety levels in children with ASD compared with typically
developing children. The sample comprised ninety-nine children; 33 with ASD,
33 with PDD-NOS and 33 typical children. The Autism Spectrum Disorders
Diagnostic for Children and Comorbidity for Children scales were used to
measure anxiety levels and level of communication deficits. The authors found
that anxiety decreased as degree of communication deficits increased for those
with ASD compared with participants with PDD-NOS and controls. Conversely
for those with PDD-NOS, anxiety was shown to increase as communication
deficits increased compared to controls. In a further study, Davis et al (2012)
investigated the effect of communication deficits on anxiety in 735 infants and
toddlers aged between 15 -36 months. This study focused more specifically on
expressive and receptive communication deficits as potential moderators of the
relationship between ASD and anxiety in this age group. It was found that both
expressive and receptive communication skills were found to be significant
moderators of anxiety in children with ASD. Individuals with ASD and PDD-NOS
demonstrated increased anxiety as communication skills increased. This finding
is consistent with Davis et al’s (2011) finding for those with PDD-NOS. The
authors posit that this is likely to support the hypothesis that increased
understanding of anxiety and emotions, facilitated by better communication skills
is likely to give rise to elevated anxiety. These studies provide interesting
findings relevant to clinical practice, and support the hypothesis that factors
unique to ASD are implicated in anxiety in ASD.
Weak central coherence
Burnette et al (2005) investigated the role of weak central coherence (WCC) in
anxiety in children with HFA. WCC is a perceptual-cognitive style that concerns
how information is processed. In the normal population, people tend to recall
information in terms of an overall impression. However, people with autism are
said to have a ‘weak drive’ for coherence, typically focusing more on details, 34
finding it difficult to ‘see the bigger picture’, which may result in difficulties
understanding the meaning or context of a situation. The main tenet of this
theory is that detailed processing is suggestive of weak central coherence which
underlies the central disturbance in autism. The authors sought to test the WCC
hypothesis, and investigate relationships between WCC, Theory of Mind (ToM)
and social-emotional functioning. Results did not support the WCC hypothesis,
and no significant relationships were found between WCC and social-emotional
functioning. This study challenges the hypothesis that WCC is a central
disturbance in ASD, and refutes the hypothesis that WCC and ToM are
implicated in anxiety in children with ASD.
Sensory over-responsivity
Sensory over-responsivity (SOR) has been posited as a causal factor in the
development of anxiety in children with ASD. Studies relating to SOR with no
relation to psychological factors were not included in the current review. Ben-
Sasson et al (2008) used cluster analysis to group toddlers with ASD with similar
sensory profiles. Results indicated that toddlers with high frequency of sensory
symptoms, and high frequency of under or over sensory-responsivity
demonstrated higher levels of anxiety, when ASD symptom severity was
controlled for. The authors posit that hypervigilance and attentional biases
central to anxiety disorders causes attention to sensory stimuli, and difficulty
regulating negative emotion and hyperarousal, which lead to over-reaction to
sensory stimuli, and SOR. SOR then exacerbates anxiety through aversive
classical conditioning, which serves to increase hyperarousal and difficulty
regulating negative emotion.
Discussion
This review aimed to provide a critical overview and synthesis of the findings of
published studies pertaining to psychological models of anxiety in children and
adolescents with ASD. Based on the findings presented in the studies included
35
in the current review, it is clear that processes evident in typically developing
children and adolescents with anxiety disorders and those related to core
symptoms are likely to be implicated in the development and maintenance of
anxiety in this population. However, it is clear that research and thus
understanding in this area is in its infancy, as many studies reviewed were
exploratory, with few testing explanatory models of anxiety in young people with
ASD.
However, there are themes arising from the research reviewed here. Firstly,
research appears to have proceeded in two streams; studies considering
whether factors implicated in anxiety disorders in typically developing children
are present in individuals with anxiety disorders and ASD, and studies
investigating the role of core ASD features in anxiety in those with ASD. These
two lines of research seem largely independent of each other, with few studies
considering the interaction of core ASD symptoms and processes seen in
anxiety disorders among typically developing children. However, the studies
reviewed that have begun to consider the relationship between the two areas
show promise (e.g. Boulter, Freeston, South & Rodgers, 2014; Bellini, 2006a;
Ben-Sasson et al, 2008), and are likely to be more clinically relevant and
representative in terms of the conceptualisation of the interaction of ASD and
anxiety and individualised treatment planning.
There were mixed results in terms of the role of levels of cognitive and adaptive
functioning in anxiety, with some finding that intelligence did have an effect, and
others not. One study (Niditch, Varela, Kamps, Hill &Trenesha, 2012) provided
support for the relationship between social understanding and anxiety, which
represents a more nuanced approach to considering specific aspects of ability
that may mediate the development of anxiety in this population, and is a
promising avenue for future research. This may reflect differences in samples, in
that some focused solely on high functioning children, and others the full range
of diagnostic groups. Results from the current review suggested that processes
36
present in typically developing children such as intolerance of uncertainty are
likely to play a role in the development and maintenance of anxiety in children
with ASD, but that these may be augmented by core features of ASD such as
rigidity of thinking. By demonstrating the mediating role of constructs such as
IOU, this research has paved the way for further study of testable models and
related interventions. Insistence on sameness, restricted and repetitive
behaviours (RRB) and restricted or circumscribed interests were a prominent
focus of the research reviewed here, which seems to have remained an area of
research since Kanner’s (1943) original hypotheses about the links between
these concepts. Most studies found a relationship between frequency of RRB
and anxiety, but others found more specific relationships, such as symbolic
enactment of restricted interests (Spiker, Lin, Enjey, Van Dyke & Wood, 2011).
This is discussed further in terms of clinical implications in a later section.
Methodological issues and future research
It is notable that the majority of studies were correlational, meaning causation
cannot be inferred, limiting conclusions that can be drawn. Indeed, this may
reflect the embryonic nature of the research field in this area, as studies were all
conducted in the last ten years, with the majority in the last two years. Ultimately,
this highlights that there is a lack of theoretically sound, tested models of anxiety
in children and adolescents. In order for research to progress to determine the
mechanisms behind associations, more experimental methodologies and
mediation and moderation analyses must be employed. Experimental designs
could manipulate factors currently implicated in the studies outlined, such as
level of uncertainty in a situation, to test relationships with anxiety. This would
provide more robust evidence and allow causal models to be developed, and
ultimately specific, empirically derived treatment targets. Treatment studies
would lead on from such experimental manipulations. Although research has
focused on trials of treatments of anxiety shown to be efficacious in the normal
population adapted for ASD, specific treatment studies relating to the factors
identified in the current review are warranted.
37
Given the exploratory nature of the field, small scale studies based on the
underlying phenomenology of anxiety in young people with ASD such as cases
studies would be beneficial to inform the development of models specific to
anxiety in ASD. Furthermore, as research remains largely at the exploratory
stage, there is a lack of longitudinal studies. It is promising that some studies
reviewed here focused on infants with ASD, but there seems to be little research
focusing on the developmental trajectory of anxiety in ASD, particularly in terms
of how factors implicated in the development and maintenance of anxiety in this
population may change over time. Current research indicates that anxiety is
elevated in adolescents with ASD, particularly in terms of social anxiety which
most commonly develops in adolescence (Ollendick & Hirshfeld-Becker, 2002).
Longitudinal research would enable understanding of the development of
anxiety in ASD, and therefore identification of risk factors that would facilitate
early detection and treatment. Longitudinal designs would be particularly
valuable in elucidating the role of psychosocial influences such as adverse life
events, victimisation and comparisons with peers, and relationship difficulties,
which have been hypothesised by some authors as likely to play a key role in
the development of anxiety in young people with ASD (e.g. Tantam, 2000,
Storch et al, 2012).
Many studies included in the current review compared anxiety across diagnostic
groups, which now limits applicability given the recent changes to diagnostic
groups and criteria in the DSM 5 (American Psychiatric Association, 2013).
Future studies need to reflect these changes in order to be clinically relevant.
There is an obvious gap in the literature in terms of theoretically grounded
models of anxiety in ASD. Therefore, any research in this area is crucial to test
theories related to treatment of this population. Treatment studies have their
obvious value, but to continue without theoretical underpinnings is questionable,
38
and thwarts the evaluation of efficacy of treatments and the development and
refinement of theoretically derived treatment targets.
The studies included in this review employed a varied selection of measures of
anxiety. The majority of studies used behavioural, parent report measures of
anxiety, often derived from broad behavioural or symptom measures for use in
children with ASD. Others used specific, validated self-report anxiety measures
that have been used in the typically developing population, tapping into more
cognitive aspects of anxiety. However, this dichotomy between parent and self-
report, and behavioural and cognitive aspects of anxiety limits comparisons.
Furthermore, using combined subscales from behavioural measures which are
not validated threatens the validity of studies. One study (Gotham et al, 2013)
used factor analysis, which strengthens validity and would help to develop
measures specific to anxiety in ASD. There are widely recognised benefits of
using multiple informants in measuring constructs such as anxiety, which would
be recommended for future research in order to improve validity by capturing
both parent and self-report, and cognitive, behavioural and physiological aspects
of anxiety.
Clinical implications
The factors considered in the current review are important in identifying risk
factors, mediators and moderators of anxiety in this population. Furthermore,
findings offer key implications for assessment, formulation and treatment of
anxiety disorders in young people with ASD. A consistent finding across the
majority of studies discussed here is the association between restricted interests
and repetitive behaviours and anxiety. This suggests that clinicians should be
aware of repetitive behaviours as a possible indicator of elevated anxiety, and in
turn consider the impact of such behaviours on anxiety. It could be hypothesised
that young people with ASD may experience the world as unpredictable and
difficult to cope with, and therefore exhibit higher levels of insistence on
39
sameness, restricted interests, repetitive or restricted behaviours. Functional
analysis, considering the antecedents, behaviours and consequences may
further explicate the role of repetitive behaviours as a coping strategy in children
with elevated anxiety.
Clinicians should be aware of processes that are common to the development
and maintenance of anxiety disorders in typically developing young people, but
be mindful that these models will not be sufficient in providing a complete
account of the person’s difficulties, to avoid a ‘one size fits all’ philosophy in
terms of treatment. It is important to note that research regarding attentional
biases to threat information, a well-established phenomenon in typically
developing children and adolescents is contradictory. This has implications for
treatments such as attentional training and cognitive restructuring, used across
many anxiety disorders, as young people with ASD may not hold these
information processing biases. Clinicians should therefore be aware of the
complex interplay between ASD core features and the psychopathology of
anxiety in young people, without overlooking one or the other. The current
research picture points to the importance of both areas, which should be
considered in the detection, diagnosis, assessment, formulation and treatment
of anxiety in young people with ASD. An emphasis should be placed not only on
individualised adaptations to current treatment approaches, but detailed
individualised assessment and formulation of anxiety in ASD, based on
presenting phenomenology. This is of particular relevance to current service
developments in UK mental health services, which are advocating reasonable
adjustments that should be made by mental health services to enable people
with autism to have equal access and effective treatment.
40
Conclusions
This study has synthesised literature relating to factors implicated in anxiety in
children and adolescents. Research appears to be proceeding in terms of
examining processes observed in relation to anxiety in typically developing
children, those related to core ASD symptoms, and those that considered the
interaction of normal processes and ASD features. Studies in all areas show
promise, particularly those relating to intolerance of uncertainty, repetitive
behaviours and restricted interests and the impact of social-communication
deficits. Research on theoretically robust psychological models of anxiety in
children with ASD is however in its infancy, and further research seeking to
replicate findings and test explanatory models is necessary, supported by
smaller scale research further exploring the phenomenology of anxiety in this
population.
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56
Service improvement project
Parents’ experiences of emotional and psychological support up to 2 years following their child’s diagnosis of a life-limiting condition
Emily Louise Howe
Doctoral Programme in Clinical Psychology, Department of Psychology,
University of Bath, Claverton Down, Bath, BA2 7AY, Tel: 01225 385506, Email:
Word count: 5038
May 2014
External/field Supervisor: Dr Jackie MacCallam, LifeTime Service, Child
Health Department, Bath NHS House, Newbridge Hill, Bath, BA1 3QE. Tel:
01225 731624, Email: [email protected]
Internal/academic Supervisor: Dr Claire Lomax, Doctoral Programme in
Clinical Psychology, Department of Psychology, University of Bath,
Bath, BA2 7AY. Tel: 01225 385091, Email: [email protected]
Proposed journal: Clinical Practice in Pediatric Psychology. This journal
publishes peer-reviewed papers representing the professional and applied
activities of paediatric psychology, including service development.
57
Introduction
Life-limiting and life-threatening conditions
Life-limiting conditions (LLC) in children describe diseases with no reasonable
hope of cure that will ultimately lead to premature death (Sutherland et al, 1993).
Life-threatening conditions (LTC) are those for which curative treatment may be
successful but carry a substantial risk of mortality in childhood (Association of
Children's Palliative Care, 2004). There are over three hundred conditions which
fall under these definitions. National prevalence of life-limiting conditions in
children (aged 0-19 years) in England have increased over ten years from 25 to
32 per 10 000 population (Fraser et al, 2012). Only recently have these
conditions been recognised as a separate category. Research is lacking on the
psychosocial needs of these children and their families. Adult palliative care has
been the focus of much of the research into the psychological impact of life-
limiting/life-threatening conditions and their treatment, with significantly less
attention being paid to children and their families with complex needs (Wray,
Lindsay, Crozier, Andrews & Leeson, 2013).
Policy context and service provision
The UK Department of Health strategy for children’s palliative care: ‘Better Care:
Better Lives’ (Lewis, 2008) and ‘Aiming High for Disabled Children’ (Department
of Health, 2008) focused on improving the outcomes and experiences of young
people and their families living with LTC/LLC. It called for the development of a
better understanding of local population needs and universal provision of
specialist emotional, psychological support for the family. The Association for
Children’s Palliative Care (ACT) Children’s Hospices UK (CHUK) emphasised
the importance of holistic family centred care (ACT/CHUK, 2009). Emotional
support within such services is recognised as the shared responsibility of all
professionals (Crawford, 2004)
58
Psychological impact
Having a child with a life-limiting or life-threatening illness can exert a
considerable impact on parents’ wellbeing (Brehaut et al, 2011; Rodriguez &
King, 2009). A recent systematic review concluded that emotional distress (e.g.
anxiety, depression) is greater among parents of children newly diagnosed with
a LTC/LLC condition compared with the normal population (Cousino & Hazen,
2013). “Chronic sorrow” is a grief reaction commonly experienced by parents
associated with “ongoing living loss that is permanent, progressive, recurring,
and cyclic in nature” (Gordon, 2009, p.115). Longitudinal studies show that
emotional distress remains high up to one year after diagnosis, and parents may
continue to experience loneliness, uncertainty, symptoms of post-traumatic
stress and fear of relapse (see Cousino & Hazen, 2013, for a review). There is
therefore a need for appropriate emotional and psychological support for
parents. However, unmet psychological and emotional support needs have been
widely reported (Beresford, 1995; Townsley et al, 2004).
It has also been noted that research is needed to explore family perceptions of
psychological need (Wray, Lindsay, Crozier, Andrews & Leeson, 2013).
Qualitative research methods have been recommended as particularly valuable
in this field in in identifying families’ needs (Emond and Eaton, 2003). Wray,
Lindsay, Crozier and Leeson (2013) explored family and stakeholder
perceptions of psychological services in a UK children’s hospice which provides
both in-house and community services in a rural area. Focus groups and
interviews were conducted with 45 parents, 95 hospice staff and 28 external
staff. Verbatim transcripts were analysed using thematic coding, which identified
two overarching themes; ‘understanding psychological support’ and ‘unmet
psychological need’. Subthemes comprised choice, staff roles and labels,
communication and flexibility. There was a lack of understanding about what
constitutes psychological support and which clinicians might provide it,
59
representing the challenge in articulating the role of psychology within
multidisciplinary care in this setting.
Service improvement needs: The Lifetime Service
The Lifetime Service is a specialist service provided by Sirona Care and Health
that was set up in 1993 to meet the complex needs of such families in Bath and
North East Somerset, UK. It comprises a multidisciplinary team of Community
Children’s Nurses, Clinical Psychologists and Health Care Support Workers.
The service has contact with up to 400 families a year who have children with a
non-malignant, LTC/LLC who may require complex health care.
In accordance with NICE guidelines for supportive and palliative care (NICE,
2004), all professionals at the Lifetime Service offer psychosocial support to all
family members at all times during the child's and family's journey, at Levels 1-3
by nurses and healthcare assistants, and specialist interventions at level 4
provided by Clinical Psychologists. However, the accessibility and perceptions of
psychosocial support is identified as a pertinent area for research. A key driver
for this research has been the services’ observation that parents’ help seeking
for psychological difficulties such as trauma has often been at a late stage,
months or years following diagnosis.
Due to the multidisciplinary nature of service provision, identification of
psychological needs and appropriate support and information provision can
prove difficult. It has been noted that parents often seek help for practical
elements of their children’s care, but may find it more difficult to seek support for
their psychological and emotional needs (Lewis, 1999).
60
Aims
The purpose of this study is to explore parents’ experiences of emotional
support following their child’s diagnosis of a life-limiting or life-threatening
condition. It aims to understand what information parents have received about
common emotional reactions and offer or provision of emotional support. We
also want to know what has been helpful about this support. This will help us to
understand parents’ needs in relation to psychological and emotional support.
Finally, we would also like to find out what services can do to improve this
support. It is hoped that the findings of the study will inform the development of
accessible written information for parents describing some of the common
emotional consequences of their child having an LTC/LLC and how they can
seek help, as well as staff training on identifying psychosocial needs particularly
in relation to information provision regarding support options.
Method
Scoping
Scoping was undertaken to assess potential research questions within the
service in terms of usefulness and feasibility. This facilitated elicitation of the
rationale for the service improvement project at the current time and needs in
terms of the nature, scope and possible outcomes of the project.
Research questions were discussed with field and academic supervisors, and a
final research design was presented to the LifeTime service multidisciplinary
team.
61
Design
A qualitative design was used due to the exploratory nature of the research. This
allowed consideration and clarification of broad and sensitive topics relevant to
the research questions such as receipt of emotional and psychological support.
Audiotaped semi-structured interviews were conducted with parents, transcribed
verbatim, and subjected to thematic analysis.
A semi-structured interview schedule was developed in collaboration with
supervisors based on previous research and service needs. The interview
structure (Appendix D) comprised the following sections:
Firstly, demographic and medical information about the child including their
health condition, time since diagnosis, number of health professionals they had
contact with and number of emergency admissions and hospital stays.
Secondly, forms of emotional and psychological support were explored,
including sources and forms of support, what was most helpful, if this was
sufficient and factors that prevented the participant from seeking support.
Furthermore, participants were asked what other forms of emotional and
psychological support would have been helpful, and at what time.
Finally, provision of information about emotional and psychological responses
was explored, including whether information about the potential impact of their
child’s condition on them, how information was received, what was helpful about
it and whether this was at the right time, if there was any other information that
would have been helpful to have received, and whether written information
about emotional impact would have been helpful, and what they think would be
helpful to include.
Participants were told that they were not required to answer every question if
they did not wish to, and were encouraged to seek clarification if they were
unsure of what was being asked. Each area of inquiry was covered. The order of
sections varied between interviews due to their similarities.
62
Participants
Participants were five parents (four mothers and one father) who were the
primary caregivers of a child or children aged under 18 with a life-limiting or life-
threatening condition. Five interviews took place with the mother alone, and one
with both mother and father of the child. Participants were included if their child
had been diagnosed six to twelve months ago, and were in receipt of active
treatment from the Lifetime service. Exclusion criteria were as follows; the child
is receiving palliative care, the participant lacks capacity to consent to
participation or Lifetime clinicians considered it detrimental to recruit a parent
guided by the clinical judgement of the team. There were no exclusion criteria
based on type of illness.
Relevant medical information about the parents’ children is summarised in table
1. Participants were recruited from service users of the Lifetime service, a
nursing and Psychology complex health care service in Bath, UK. Recruitment
was carried out by clinicians of the Lifetime service using convenience sampling.
Table 1.
Participant information
Participant Child’s condition Type of condition
Time since diagnosis
1 Not disclosed - 4 years2 Metabolic disorder LLC 18 months3 Congenital (chromosomal) disorder LLC 2 years4 Congenital Adrenal Hyperplasia LTC 4 years5 Cerebral palsy and epilepsy LLC 7 years
63
Procedure
Suitable participants were identified on the patient system by LifeTime
administration staff in accordance with inclusion and exclusion criteria. Clinicians
then sent potential participants a letter, information sheet and consent form
inviting them to participate. Informed consent was gained, and participants then
returned the consent form to the first author if they agreed to take part. The
response rate was 2.68%. Following clarification of the study requirements and
the opportunity to ask questions, an appointment was made to conduct the
interview. Interviews were conducted by the first author at participants’ homes
and adopted a non-interventionist stance. Participants were reminded that they
did not have to answer every question, that they could ask questions during the
interview and it would last approximately 40 minutes. Immediately following the
interview, participants were debriefed. Participants were asked how they found
the interview and how they were feeling. Appropriate steps were in place to
support participants should they experience significant distress. The protocol of
the current study was approved by the University of Bath Ethics Committee, and
did not require NHS ethical approval.
Analysis
Audio recordings of the four interviews were transcribed verbatim by the first
author. Transcripts were analysed in accordance with the established
conventions of theoretical thematic analysis (Braun & Clarke, 2006) to identify,
examine and record patterns or ‘themes’ within the data. The first author read
and re-read each transcript several times, making notes at the side of each one
to identify themes for coding. Codes were ascribed to each pattern or theme.
Coded data were collated into meaningful clusters, and used to generate
overarching themes. Subthemes were then identified which were closely related
to the original data. Analysis involved continued reference to the transcripts,
extracts and themes in order to ensure that themes were grounded in the
original data. Overarching themes were developed according to the research
64
questions and clinical implications for service improvement. Finally, transcripts
were re-read to ensure that the themes fitted the original transcripts.
Results
Two overarching themes and nine subthemes were identified from thematic
analysis of the transcripts. These are presented in Table 2. The first theme,
‘psychological and emotional support’ is comprised of a number of subordinate
themes related to parents’ experiences of psychological and emotional support
and their needs in relation to this. The second theme, ‘psychological distress’
taps into participants’ experiences of distress and the responses to this. In the
next section, each overarching theme and its subordinate themes are described.
Direct quotations are presented alongside discussion to illustrate the themes.
65
Table 2
Overarching themes and sub-themes
Psychological and emotional support
- Prioritising the child’s needs over your own
- Lack of support
- Timeliness of support
- Understanding the psychological support available
- Access to psychological support
- Support needs
Psychological distress
- Experience of psychological distress
- Knowing what’s normal
- Stigma
Psychological and emotional support
This broad theme captures parents’ experiences of seeking, understanding and
receiving psychological support from the Lifetime service and services prior to
this. It also relates to parents support needs.
Prioritising the child’s needs over your own
Three participants described putting their child’s support needs before their own,
perhaps reflecting the intensive support that their children need. When talking
about what stopped her taking up an offer of psychological support soon after
diagnosis, Participant 1 said:
66
[Child’s] illness is horrible and sad and obviously, at times you need
support, but, in that first instance, I just needed to make sure everything
was as good as it could be for the outcomes, and that was my goal
Participant 1 expanded on this in commenting:
Because you’re sort of secondary to it. So, for yourself, you’re just living
day by day, rather than… I mean, obviously, you’re supposed to take
care of yourself because you’re no good to anybody without it, but your
priority isn’t yourself really.
Another participant talked about their own life in general being secondary to their
child’s:
Participant 2: So you kind of put your life on the backburner and
everything goes…
Finally, participant 3 also referred to support being focused around their child:
…the fact that they’re there to help with, to be there for him, it’s sort of
we’re just on the side-lines really.
Lack of support
This subordinate theme relates to two participants’ experiences of lack of
support following diagnosis, before they came into contact with the Lifetime
service. Participant 1 referred to a lack of support from services:
…for over a year, we were basically in the wilderness, not getting any
kind of support really. So, I think now we have all the support we need,
but previous to that…
…until that moment, there was just a big void
67
When asked about emotional support from family and friends, participant 1
described friends and family finding it difficult to respond to the family’s situation,
which she believed was due to their own reactions to the child’s diagnosis:
Of sorts. I think, because nobody… people need to seem to need to
understand the situation to help support but, because nobody had heard
of it, nobody knew. People don’t seem to know how to say things or what
to do because they don’t know what it is and I think, when there’s
undiagnosed or unknown… what she’s got is not widely known of, so
people don’t know how to respond to it. They can be quite freaked out by
it.
Participant 2 described never being offered psychological or emotional support:
So no, never really been offered it
Timeliness of support
Participants described wanting information, or the offer of support early on
following diagnosis. Participant 1 said:
… you know, you’re probably not ready for a service the day you find out,
you’re diagnosed. That’s probably just a bit too much but somewhere
between then, a little bit further down the track, information needs to
come into you that helps support the family, which didn’t at that point.
She went on to say:
Yeah, I think just really getting that support from near on day one. You’re
not ready to absorb it on day one, but to say to someone something
bad’s… you know, whatever the diagnosis is and then say, “We’ve hit you
with enough for today but perhaps, in a couple of weeks’ time, we can call
you and give you access to other agencies”.
68
…after about a couple of weeks, it’s really hit you I think, you know, what
the situation is and I think, any time between two weeks and a month, I
think you need to know..
It should come from diagnosis. It should set off a chain reaction of
support really.
Participant 5 commented:
…I do think support’s missing though for when people are initially
diagnosed.
Because you kind of get there yourself, I suppose, eventually but I think
they need it more early on.
Commenting on whether the leaflet from the hospital detailing different support
options was provided at the right time, participant 2 said:
I don’t know really. I think the shock from the diagnosis, I think maybe if
they sent it in the post a week later or someone touched base and
phoned up and gone…
Participant 4 described being satisfied with the support she had received from a
Psychologist at the Lifetime service, but would have liked this earlier in her
child’s care:
I would have liked more, earlier. Yeah, I think especially sort of after
diagnosis I mean his consultant was great, but as a family it’s an awful lot
to take on and to come to terms with. And I think if we could have had
that, from the start, or from three months, or six months, it would have
made a huge difference.
69
Understanding psychological support and how to access it
Participants referred to wanting information about what psychological support is
on offer and how to access it:
Participant 1: I think you kind of need like a bible of services, really, and
just a little description of what they do and what they’re about and what
their remit is
Participant 1 went on to describe what would be helpful in relation to information
about emotional responses to caring for their child and support available:
…I think recognising the responses and knowing where to go to if you
recognise those responses in yourself, is just perfect really
So it would be beneficial to sort of have maybe like a warning sign, kind
of…
.…if you’re feeling like this, it might be time to just give someone a quick
tinkle, yeah.
You see I would just… if I had these booklets, I’d put them in the
cupboard and read them at my leisure.
Participant 2 described wanting to know who to contact if she wanted
psychological support from the service:
I don’t know because we’ve never been offered a counsellor and, if I
wanted one, I would have no idea about what route to go down. Would I
have to go to the doctor and say, “I need a counsellor”?
Even if I wanted one, I wouldn’t know who to phone. So if I want to talk to
somebody, what do I do?
Participant 3 had found it helpful to have easily accessible support:
70
…it is easier for me to ask someone who is already in my home, or when
I go to the hospital.
Support needs
In terms of psychological and emotional support needs, two participants felt that
simply knowing the support was available from the service and how to access it
would be sufficient:
Participant 1: I think recognising the responses and knowing where to go
to if you recognise those responses in yourself, is just perfect really.
…And then whatever comes from that would be tailored to the individual
in itself, but just knowing those signals of potential problems or however
you label it and knowing who’s your contact, who’s that person, what they
do, you know, what their role is, what the process is.
Participant 2: It’s having the choice as a parent that the service is there if
you need it, and I think that’s what we need, that there’s something there.
Participant 1 felt that tailored support was important:
I think they look at your personality and your situation and I think they
tailor what they offer in the first instance to you and the family and the
situation. So I think they probably come in, “Okay, she needs to know
that there’s a place and a safety net and something, but she’s not ready
to go down further…”
and provision of information for fathers:
I think possibly something for the dads
…it does seem very focused on the female of the house
71
…and there’s sort of nothing really for men. If you could put like a pack of
warning signs for a man, which is a bit more difficult, because they’re less
open to feelings, but if there is ever anything that could be written in a
manly kind of…
Participant 4 commented on how helpful input from a psychologist at the
Lifetime service had been for her husband (the father of their child) and them as
a couple:
My husband was kind of really you know sceptical at the start he thought
it was something would really benefit me and was happy to partake. But
at the end of it he was kind of you know I think I might have got more out
of that than you did even. So, it was really helpful, really useful you know
for both of us.
Participant 4 also described what was helpful about this more generally:
…and then the psychological support. Again, it’s fantastic because you
have all these sort of fears, and worries, but then to have like a structured
way to work through some of them, was really helpful.
Experience of psychological distress
Participants did not go into detail about their experiences of psychological
distress. However, two participants made reference to their experiences.
Participant 1 described her experience that prompted organisation of support
services such as respite:
Participant 1: …although the doctors did it all by me sort of having a mini
breakdown…
Participant 2 simply described the experience of caring for her child:
72
I realised our life is not the same like other families, but it is stressful, very
stressful I think.
Knowing what’s normal
Two participants described wanting to know what’s ‘normal’ in terms of
emotional responses to their child’s diagnosis and ongoing treatment or care,
emphasising the importance of normalisation. Commenting on whether it would
be helpful to receive information on common emotional responses in parents,
Participant 1 said:
Yeah, I think that’s quite interesting to have because you know then that
your reactions or your responses are not unique to you. It’s sort of a
commonplace.
And these responses are things that would be normal or expected and
so, by knowing you’re acting in a normal manner, you could sort of take
the pressure off yourself, can’t you, I think?
Participant 2 expressed similar thoughts:
…you don’t know how you’re meant to feel in certain situations but, if
you’re feeling like days like, “It’s not fair, why did this happen to us?” Is
that a normal response or not? And it’s nice maybe to be able to gauge
that against something a professional has done and then you don’t
maybe feel so, yeah, worried about it.
Participant 5 expressed that it was important to hear experiences of other
parents in relation to emotional experiences:
…because then you know people do feel like that…
You know, someone else who has been through it to say, you know, don’t
worry it will be alright.
73
Participant 4 however held a different view, feeling that information about
common responses would not make a difference:
You know how you’re feeling. You know that it’s probably quite normal to
feel that way, but it actually doesn’t really make a difference.
Stigma
When referring to people talking about seeing a Psychologist, participant 1
talked about responses to this, perhaps about the stigma associated with seeing
a Psychologist:
…people freak out
Participant 2 described her experience of barriers to seeking psychological
support from her GP, reflecting perhaps the stigma involved and a lack of
understanding of psychological support:
Would I have to go to the doctor and say, “I need a counsellor”? And
then I would be worried that he would think that I was losing the plot and
therefore not a fit mum to look after my child.
Discussion
This study sought to explore parents’ experiences of emotional support following
their child’s diagnosis of a life-limiting or life-threatening condition. It aimed to
understand what information parents have received about common emotional
reactions and offer or provision of emotional support, and understand what has
been helpful about this support.
74
Results highlighted similar themes to that found by Wray, Lindsay, Crozier,
Andrews & Leeson (2013), in terms of parents understanding what
psychological support is available, what it involves and how it can be accessed.
Timeliness of this support was a pertinent issue, with two participants
highlighting a preference for early information provision and support. Notably,
most participants felt that knowing that services are available and how to access
them was sufficient.
A theme of parents prioritising the child’s needs over their own was evident. This
may reflect the parents’ focus on the treatment and care of their child, which is
often intensive and ceaseless, requiring a great deal of practical and emotional
support required on the part of parents. This has been observed in previous
studies (e.g. Whiting, 2013).
A lack of support between diagnosis and referral to the Lifetime service was
experienced by some participants. This does not relate directly to provision of
psychological and emotional support by the Lifetime service. However, it would
be of value to consider Psychologists within Lifetime providing training and or
consultation to clinicians and services involved in diagnosis and early care
provision on emotional and psychological support at Levels 1-3 of the NICE
guidelines (NICE, 2004).
A theme of ‘knowing what’s normal’ arose from two participants’ experiences of
psychological distress. Both felt that it would be helpful to receive information
about parents’ common emotional responses to caring for their child, one
reflecting that this would take the pressure off and provide a sense of knowing
that these experiences are commonplace. Normalisation appeared to be
important to providing reassurance for parents, and also knowing when to seek
help according to common signs of psychological distress. Common emotions
and experiences of parents’, possibly including descriptions provided by parents
who have received or are currently receiving support from the Lifetime service
could be included in an information leaflet about parents’ experiences and
support. Furthermore, training of other professionals could include skills
75
development on helping to normalise parents’ experiences, and identifying signs
that onward referral to a Psychologist would be indicated.
It was illuminating that some participants did not know that psychological
support was available through the Lifetime service, and did not know how to
access it should they need it. Along with putting the child’s needs before their
own, and stigma associated by some with accessing support, this may provide
some explanation for why clinicians have observed parents seeking help long
after diagnosis. Similarly to Wray, Lindsay, Crozier, Andrews & Leeson’s (2013)
findings, this highlights a need for parents to be provided with information
regarding what psychological support is available and who should provide it.
This could be included in an information leaflet for parents, detailing not only
how to access formal psychological support from Lifetime Psychologists, but
also the role of other staff.
The current findings will be presented to the Lifetime service at a team meeting.
The final report will also be circulated to all clinicians. The commissioners of this
project would like to use the findings of the study to inform the development of
an information leaflet for parents covering these issues, and to inform staff
training on responding to emotional needs. The information leaflet will include
information on common emotional responses at different phases of their child’s
care and signs that indicate that further help might be beneficial. Information will
be presented in a non-stigmatising way, emphasising that support is open to all,
and does not mean that parents are ‘struggling’. It also aims to clearly outline
what psychological and emotional support is available, and how this might be
tailored to the individual’s needs. The information will also refer to fathers’
needs.
In terms of building upon the findings of this study, it would be beneficial to
review clinicians’ perceptions on provision of psychological support, particularly
at Levels 1-3. This would help to understand how support is provided in practice,
and delineate staff training needs and preferences to further improve the 76
provision of psychological support and ensure that the service is adhering to
NICE guidelines (NICE, 2004). This may form part of a future service
improvement project.
Limitations
Due to difficulties in recruiting participants to the study, the sample size is
smaller than what we set out to achieve. This affects the representativeness of
participants’ views. It may be that parents felt disengaged from the service, or
were more focused on their child’s needs to participate in a study that was
focused on their own, a theme that was reflected in the results as a barrier to
seeking support. This may highlight that the service needs to focus on promoting
the presence of Psychology within the Lifetime service in order to increase
accessibility.
More pro-active recruitment methods such as engaging with team members to
recruit via clinicians, or snowball sampling may have increased participant
numbers. In addition, it was noted that three out of the four participants had not
received psychological support from a Psychologist within the team. It could be
argued that the sample should have comprised an equal number of those who
had and had not received formal psychological support in order to explore views
of more informal support, and accessing Psychology, and views of formal
psychological support received. However, it proved helpful to hear the views of
parents who had not sought support, and barriers to this in terms of lack of
information and understanding of psychological support.
A further limitation of this study is that it did not address father’s needs. A recent
review of the gender imbalance in research concerning parental perspectives in
paediatric palliative care, concluded that research does not equally reflect the
experiences and needs of mothers and fathers (Macdonald, Chilibeck, Affleck
and Cadell, 2010). Gender differences are relevant to parenting a child with an
illness (e.g. Pelchat, Lefebvre & Levert, 2007). Results of the present study
should be interpreted with caution.
77
In terms of the short time frame for the project, it was not practicable for results
to be shared and verified with participants. However, a summary of the final
report will be made available to participants. This study focused specifically on
the needs of parents of children with LTC/LLC.
Finally, transcripts were analysed by a single author. Investigator triangulation,
whereby another researcher examines the data with the same method may have
increased the validity of the findings, and developed a broader and deeper
understanding of the issues.
Conclusion
There were a number of unmet needs identified in terms of the LifeTime
services’ provision of psychological support and information. Parents highlighted
a need relating to understanding what psychological support is and how to
access it. Many parents felt that early support and information was important
(soon after diagnosis of their child). They felt that knowing what services were
available and how to access them would be helpful. Parents also wanted to
know what emotions were ‘normal’ to experience in response to their child’s
diagnosis and treatment, and when they might want to seek help. The study has
highlighted that it would be beneficial to provide early emotional support for
parents following diagnosis. As the Lifetime service is not often involved at the
diagnosis stage, it would be important for Lifetime professionals to provide
advice and support to professionals involved with the family about emotional
support. Consideration of fathers’ needs is a key priority. Finally, it would be
helpful to find out about staffs’ views on providing emotional support in their
roles. This would help determine how support is provided in practice and
whether the service is following national guidelines (e.g. NICE, 2004).
78
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Wray, J., Lindsay, B., Crozier, K., Andrews, L. & Leeson, J. (2013). Exploring
perceptions of psychological services in a children’s hospice in the United
Kingdom. Palliative and Supportive Care, 11, 373-382.
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Lay summary
Parents’ experiences of emotional and psychological support up to 2 years following their child’s diagnosis of a life-limiting condition
Life-limiting and life-threatening conditions in children
Life-limiting conditions describe diseases with no reasonable hope of cure that
will lead to premature death (Sutherland et al, 1993). Life-threatening conditions
are those for which curative treatment may be successful but carry a substantial
risk of death in childhood (Association of Children's Palliative Care, 2004). There
are over three hundred conditions which fall under these categories.
The number of children living with these conditions in England has increased in
the past 10 years and continues to rise (Fraser et al, 2012). Only recently has
research considered the psychological and social needs of these children and
their families.
We know that when a child receives a diagnosis of a life-limiting illness this can
be an emotional experience for parents. It is common for parents to feel
distressed, or experience anxiety or depression (Cousino & Hazen, 2013).
What the study is about
In this study, we wanted to explore the experiences of emotional support parents
received following their child’s diagnosis of a life-limiting condition. It aimed to
explore what information parents have received about common emotional
reactions and what emotional support parents have received. We also want to
know what has been helpful about this. Finally, we wanted to find out what
services can do to improve this support.
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What we did
We interviewed five parents of children with a life-limiting or life-threatening
illness who were receiving help from the Lifetime service. The Lifetime service is
a health care team in Bath, UK for children with complex physical health needs.
The interview involved a set of questions about parents’ experiences of
emotional support and information about emotional responses in relation to their
child and their diagnosis.
Interviews were roughly 30 minutes long and were audio recorded and put into
written form. The written records were read and analysed to identify common
themes in what parents had said.
What we found
There were two main themes identified: psychological and emotional support
and psychological distress. Parents wanted to understand what psychological
support was and how people can access it. Many parents felt that early support
and information was important (soon after diagnosis of their child). They felt that
knowing what services were available and how to access them would be helpful.
Parents also wanted to know what emotions were ‘normal’ to experience in
response to their child’s diagnosis and treatment, and when they might want to
seek help.
Only five parents took part in the research, and this did not include fathers.
Therefore further research is needed with more parents, particularly fathers.
84
What it means for the Lifetime service
The results of this study will be presented to the team at the Lifetime service,
and in staff training.
The study has highlighted that it would be beneficial to provide early emotional
support for parents following diagnosis. As the Lifetime service is not often
involved at the diagnosis stage, it would be important for Lifetime professionals
to provide advice and support to professionals involved with the family about
emotional support.
It is important to let parents know what types of psychological support are
available through the nurses and Psychologists. Information on common
emotional responses at different phases of their child’s care and signs that
indicate that further help might be beneficial. This could be provided in an
information leaflet.
It would be helpful to find out about staffs’ views on providing emotional support
in their roles. This would help us understand how support is given in practice
and whether the service is following national guidelines (e.g. NICE, 2004). This
would help us to improve support at the Lifetime service.
85
Predictors of change in health anxiety, quality of life and psychological distress in UK cancer patients in remission: A prospective study
Emily Louise Howe
Doctoral Programme in Clinical Psychology, Department of Psychology,
University of Bath, Claverton Down, Bath, BA2 7AY, Tel: 01225 385506, Email:
Word count: 5650
September 2014
Internal/academic Supervisor: Professor Paul Salkovskis, Department of
Psychology, 6 West 0.9, University of Bath, Claverton Down, Bath, BA2 7AY
Tel: 01225 384350 Email: [email protected]
External/field Supervisor: Dr Anna Lagerdahl, Great Western Hospitals NHS
Foundation Trust, Dove Unit - 3rd Floor, Great Western Hospital, Swindon, SN3
6BB
Tel: 01793605323, Email: [email protected]
Proposed journal: Journal of Cancer Survivorship. This is a peer reviewed
journal that publishes research that improves understanding of survivorship
topics affecting quality of care and quality of life. Recent papers focus on fear of
cancer recurrence.
86
Introduction
The transition from the treatment phase of cancer to remission can be a difficult
time for cancer survivors. Evidence suggests that though most people adjust
well following treatment with curative intent, a significant proportion develop
psychological difficulties, leading to poorer quality of life (QoL) [1-3]. Indeed,
research into the experiences of cancer survivors has shown that these
psychological difficulties are more of a priority than physical problems [4]. This is
reflected in the recent focus on improving psychological care for this population
[5].
Typically, researchers have approached psychological adjustment in cancer
survivorship by determining rates and correlates of broad psychiatric diagnoses
such as anxiety and depression, derived in mental health populations. Anxiety in
particular has been shown to be elevated in cancer survivors [e.g. 6-8].
However, applying such diagnoses in the context of cancer survivorship may not
be a particularly helpful or clinically meaningful way to understand distress as a
reaction to potentially life-threatening illness; being distressed in such
circumstances is the rule, not the exception, making diagnoses close to
meaningless. A more nuanced psychological approach is warranted in order to
understand the ways in which psychological variables mediate the impact of
serious physical illness. By implication, this could suggest possible general and
more targeted interventions.
At present, research relating to possible reactions to the experience of cancer
predominantly focuses on non-psychological aspects such as medical and
treatment factors, as well as anxiety and depression. Therefore, it seems
appropriate to refer to possible reactions to cancer diagnosis and treatment from
a more psychological standpoint, particularly in terms of concepts related to
“catastrophizing” and loss. We will summarise the rather sparse literature on
both specific and general psychological variables relevant to predicting distress
and QoL in cancer survivors, going beyond the concepts of anxiety and mood
problems where appropriate. Psychological constructs likely to interact with
87
medical conditions include those conceptually connected with anxiety. Fear of
Cancer Recurrence (FCR) is common in cancer survivors [9] and is related to
health anxiety observed in other medical and non-medical populations [10]
including cancer patients [11-14]. Both constructs exist on a continuum, from
minimal to significant and excessive. Intolerance of uncertainty (IOU) is a
construct that has been well researched in the development and maintenance of
anxiety, and more recently in relation to health anxiety in cancer survivors [15].
Linked to but probably separate from low mood, Mental Defeat is a construct
similar to demoralisation and depression as commonly experienced by cancer
survivors, which has been shown to predict levels of anxiety and low mood in
patients with pain; it could be argued that it corresponds to catastrophizing in the
domains of identity and perception of social role [16]. Existential concerns have
also been noted in cancer survivors and have been found to be associated with
lower QoL and wellbeing [17]. At a more metacognitive level, unhelpful beliefs
about the experience and expression of emotions in general have been
implicated in the maintenance of distress in a number of conditions, including
physical health problems such as chronic fatigue syndrome [18].
Catastrophizing, which can be defined as dwelling on the worst possible
outcome of any situation in which there is a possibility of an unpleasant outcome
[19, 20] is a cognitive process that is key to health anxiety and FCR. It could
also be argued that mental defeat is also a form of self-catastrophizing [16].
These psychological variables will be explored in more detail in the following
sections.
Fear of recurrence
It is not surprising that those who have undergone treatment for cancer often
continue to be preoccupied by whether or not the problem is entirely resolved, at
its most extreme manifesting as a catastrophizing response. Fear of cancer
recurrence (FCR) has been defined as the fear that cancer will recur, progress
88
or spread in the same or a different part of the body [21]. A prevalence rate of
moderate-to-high FCR has been reported at 42% in cancer survivors [9]. FCR is
a normal and understandable response to the threat of cancer recurrence, but
can become excessive and persistent even when risk of recurrence is low [22]
predicting poorer QoL up to 11 years post diagnosis [21, 23]. There is an overall
indication that psychological factors are more highly associated with FCR than
clinical factors such as cancer site or stage [23].
Models of fear of recurrence and health anxiety
Authors have noted the similarity of cognitive and behavioural processes
involved in maintenance of FCR and health anxiety [6, 24, 25]. Health anxiety is
defined as intense and persistent anxiety about one’s present and future health
linked to catastrophizing interpretations [26]; it has been shown to be elevated in
medical populations [e.g. 10, 27]. Lee-Jones’ [24] original cognitive model
posited that FCR leads to heightened awareness of and hypervigilance for
internal and external cues such as bodily sensations. These cues are
misattributed to cancer, which can trigger significant distress. Safety seeking
behaviours [28] such as reassurance seeking (particularly from inappropriate
sources) serve to increase perceptions of threat and thus FCR. Severity of FCR
has been shown to predict frequency of reassurance seeking and healthcare
usage [29, 30].
Similarly, the cognitive model of health anxiety [26] proposes that people with
elevated health anxiety tend to misinterpret ambiguous physical sensations and
other health information as evidence that they currently have or are at
exceptional risk of developing a serious illness or illnesses [31]. Related
attentional, physiological and behavioural processes serve to maintain the
person’s sense of threat, and further increase anxiety. In terms of the
development of health anxiety, idiosyncratic fears are thought to arise from
personal or family experience of a distressing illness or treatment, which lead to
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specific health related assumptions [31]. This model of health anxiety was
derived in relation to people without medical conditions. However, it is now
known that health anxiety is common in people who are currently physically ill,
or where there is reason for them to consider their longer-term health status as
ambiguous. Similarly to FCR, health anxiety exists on a continuum, from minimal
to excessive and clinically significant [31]. The model has been shown to apply
in general medical settings [32, 33] and in people with cancer, diabetes, multiple
sclerosis and chronic pain [10, 13, 14, 27, 34, 35].
To date, four studies have investigated health anxiety in cancer patients, solely
in relation to breast cancer [11-14]. However, it is not clear from these studies
what proportion of samples had completed treatment with curative intent, and
what proportion were still undergoing treatment. In studies that used measures
of health anxiety validated for in medical populations increased anxiety
sensitivity, greater bodily vigilance and poor social support were significantly
associated with elevated health anxiety [13, 14].
Mental Defeat
In terms of the more dysphoric forms of psychological distress, cancer survivors
have been found to experience demoralisation; characterised by hopelessness,
helplessness and a loss of meaning and purpose [36, 37]. Mental defeat is a
relatively new concept recently studied in patients with chronic pain,
characterised as a type of self-catastrophizing [16, 38]. Mental defeat is
characterised by negative thoughts and beliefs around loss of autonomy
affecting the person’s identity, agency and self, and was derived from models of
depression [39] and Post Traumatic Stress Disorder [40], both relevant in cancer
[41-43]. Mental defeat has been found to be strongly associated with anxiety,
depression, functional disability and psychosocial disability in patients with pain
[16].
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Existential concerns
Research into the existential experiences of cancer patients suggests that
issues around fear of death, uncertainty, vulnerability, isolation and loss of
meaning, among others, are common [17]. These concerns may impact
negatively on wellbeing and QoL if unresolved [17]. A recent qualitative study of
the existential experiences of cancer patients in remission found that the post-
treatment phase may serve as a catalyst for the existential concerns. Patients
are thought to become more aware of their mortality and wish to live a more
meaningful life, but find this challenging [44].
Beliefs about emotions
Unhelpful beliefs about the unacceptability of experiencing or expressing
negative emotions have been found in people with Chronic Fatigue Syndrome
[18], and mental health problems. These beliefs may lead to attempt to over-
control emotions, and have been shown to predict maintenance of psychological
distress.
Intolerance of uncertainty
Intolerance of uncertainty (IOU) is defined as a cognitive bias that affects how a
person perceives, interprets, and responds to uncertain situations [45, 46] and
has been shown to moderate the relationship between catastrophic health
misappraisals and health anxiety in people with high levels of health anxiety
[47]. Cancer patients are inevitably exposed to some level of uncertainty in
terms of recurrence. Studies in lung and prostate cancer patients have shown
higher levels of IOU to be related to poorer emotional wellbeing and cancer
related distress up to 5 years post-diagnosis [15, 43].
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Aims
Cognitive behavioural therapy (CBT) has been shown to be an efficacious
treatment for health anxiety physical health settings [33]. Combined CBT and
health psychology approaches are promising for FCR [48, 49]. However, little
research has investigated health anxiety or the specific psychological predictors
of psychological outcomes in cancer survivors. The present study seeks to
contribute to research in this area by investigating whether (a) mental defeat, (b)
existential concerns, (c) beliefs about emotions, (d) and intolerance of
uncertainty; can predict levels of (e) health anxiety (pre-occupancy and worry
that one has a serious illness), (f) quality of life and (g) psychological distress in
cancer patients who are in remission (i.e. have completed treatment that has
cured the disease), and to investigate change over time.
Method
Design
A prospective questionnaire design was employed with data collected at two
time points (T1 and T2), four weeks apart. The study comprised four predictors;
mental defeat, existential concerns, beliefs about emotions and intolerance of
uncertainty and three outcomes; primarily health anxiety secondarily quality of
life and as a tertiary variable, psychological distress.
Participants
Ninety participants aged 23-80, who had completed treatment for cancer with
curative intent, were recruited using convenience sampling from oncology and
haematology departments at two UK hospitals, three support groups and the
Macmillan cancer voices website. Sample size was based on an a priori power
calculation using G* Power [50] which computed a total sample size of 85, with 4
predictors, a medium effect size and 0.8 power, based on current literature (e.g.
51]. This level of power was achieved (power = 0.83).92
Participants were eligible if they had been diagnosed with and undergone
cancer treatment with curative intent, had been declared clear of the disease
were currently in receipt of care from their treating hospital, aged 18-80 years
and were sufficiently fluent in English to be able to understand and complete the
questionnaires. Participants were excluded if they lacked capacity to give
informed consent to participate, or had a recurrence of cancer during the study.
Suitable participants were recruited by Consultants and Clinical Nurse
Specialists in oncology or haematology, or participants self-selected by
responding to an advert in the case of the support groups and the Macmillan
cancer voices website. Informed consent was gained in written, signed form.
Flow of participants through the study is summarised in Figure 1, overleaf.
93
Figure 1.
Flow diagram of participants through the study.
Measures
Participants were asked to complete a questionnaire pack (Appendix E). They
were asked to provide demographic information regarding their age, sex, type of
cancer, type(s) of treatment, time since completion of treatment, health
94
Excluded
Did not meet inclusion criteria (n = 10)
(Non UK resident: n = 1, cancer recurrence: n =7, age: n = 2) Outlier (n=1; time since treatment = 21.8 years)
Excluded Cancer recurrence (n = 2)
Demographic data not completed (n = 2)
Lost to follow up (n = 17)
CompletedT1 questionnaires
(n = 90)
Assessed for eligibility(n = 119)
CompletedT2 questionnaires
(n = 69)
Did not return T1 questionnaires
(n = 18)
conditions and dates of their previous and next follow up appointment with their
Consultant Oncologist or Haematologist.
The following self-report questionnaires were used to collect the data at both
time points. A full description of each measure is included in Appendix F. They
were piloted with a person with personal experience of cancer to assess
approximate time taken to complete questionnaires and acceptability.
Mental defeat
The Pain Self Perception Scale (PSPS) [52] is a 24 item scale adapted from the
Defeat Scale [39] and Mental Defeat during Trauma Scale [53]. Items are rated
from 0-4 giving a range of 0-96. The PSPS was modified for the present study
with permission from the authors by adding the statement: ‘Because of my
experience of cancer and treatment..’.
Existential concerns
The Life Scheme subscale of the Spirituality Index of Wellbeing (SIWB) [54] was
used to measure existential concerns. Items are rated on a 5 point scale ranging
from 1 “strongly disagree” to 5 “strongly agree”, generating a range of 6-30.
Reverse scoring was used to facilitate analysis, with higher scores indicating
greater existential concerns.
Beliefs about emotions
The Beliefs about Emotions Scale (BES) [18] is a 12-item scale that measures
beliefs about the experience and expression of emotions. Items are rated on a
scale from 0 to 6, with a range of 0-72. Higher scores indicate more maladaptive
beliefs.
95
Intolerance of uncertainty
The Intolerance of Uncertainty Scale-12 (IUS-12) [55] is a 12 item version of the
original IUS, a 27-item measure of intolerance of uncertainty [56]. Items are
rated on a scale from 1 ‘not at all characteristic of me’ to 5 ‘entirely characteristic
of me’, with a total scores ranging from 27 – 135. Higher scores indicate higher
levels of intolerance of uncertainty.
Health anxiety
The Health Anxiety Inventory Short Week (SHAI) [57] is an 18 item measure of
health anxiety. The first 14 items were used to assess levels of basic health
anxiety. The anticipated burden of illness; ‘burden’ is measured by 4 additional
items. Items are rated on a 4 point scale from 0 – 3, with a range of 0 – 42.
Higher scores indicate higher levels of health anxiety. A cut off score of 15
indicates elevated health anxiety [58] and a score of 18 or higher denotes
people who are likely to meet criteria for a diagnosable disorder [59, 60].
Avoidance and reassurance seeking subscales were also used to assess
reassurance seeking (from both non-medical and medical sources) and
avoidance behaviour comprising 10 items and 8 items, respectively.
The measure was adapted for the current study with permission from the author
to include the following statement; ‘Please answer in relation to worries about
your health in general (this may include worries about cancer)’.
Quality of life
Quality of Life Index Cancer Version III (QLI-C III) [61]. The QLI-C is a 33-item
measure of quality of life measuring satisfaction across a number of domains.
Part 1 measures how satisfied the respondent is with areas of their life on a
scale of 1 ‘very dissatisfied’ to 6 ‘very satisfied’. Part 2 measures how important
each area is to the respondent on a scale of 1 ‘very unimportant’ to 6 ‘very
96
important’, generating a range of 0-30, with higher scores indicating better
quality of life.
Psychological distress
Patient Health Questionnaire (PHQ-9) [62] and Generalised Anxiety Disorder
Assessment (GAD-7) [63] (combined: 16 items). The combined PHQ-9 and
GAD-7 were used to assess depression and anxiety as a measure of
psychological distress. The PHQ-9 total score ranges from 0 to 27 with higher
scores indicating higher levels of depression. A total score for the seven items
ranges from 0 to 21, with higher scores representing higher levels of anxiety.
Procedure
Suitable participants were identified by cancer clinicians at two UK NHS hospital
sites in accordance with inclusion and exclusion criteria. Clinicians explained the
study, gave participants an information sheet and gained consent to share their
contact details with the researcher. Participants were then contacted by the first
author to discuss the study further and answer any questions. If they agreed to
take part, participants were sent a consent form and first questionnaire pack to
return in a pre-paid envelope. Participants were offered the choice of completing
the questionnaires at home, or at their hospital with the support of the first
author if required. Participants recruited from Macmillan cancer voices
responded to an advert on their website and participants from the support
groups responded to an email circulated by the group lead. They were then
contacted by the first author in the same way as described above.
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Ethical approval
Ethical approval for this study was granted by the NRES Committee North East
– Newcastle and North Tyneside 1 Ethics Committee and the University of Bath
ethics committee. Research and development approval was granted by Great
Western Hospitals NHS Foundation Trust and Salisbury NHS Foundation Trust
(see Appendix G for documentation).
Statistical analyses
Statistical analyses were performed using the Statistical Package for the Social
Sciences (SPSS) version 21.0. An alpha level of 0.05 was used unless
otherwise specified.
Treatment of data
The following demographic and medical data was missing from the final dataset;
age (1 case), treatment type (1 case), time since treatment (3 cases), presence
of other health conditions (1 case). It is assumed that these data were missing at
random and were therefore not substituted for analyses.
An a priori decision was made to use mode substitution for missing
questionnaire data in cases where less than two items were missing due to item
non-response. At T1, this was performed for the following measures: mental
defeat (2 cases), beliefs about emotions (2 cases), intolerance of uncertainty (2
cases), and depression (as part of the measure of distress) (3 cases). At T2, this
was performed for health anxiety questions 1-14 (2 cases) and depression (1
case).
98
Tests of assumptions
Tests of assumptions for multiple regression were conducted. Tests indicated
that multicollinearity was not a concern. The data met the assumption of
independent errors. The histogram of standardised residuals indicated that the
data contained approximately normally distributed errors, as did the normal P-P
plot of standardised residuals, which showed points that were not completely on
the line, but close. The scatterplot of standardised residuals showed that the
data met the assumptions of homogeneity of variance and linearity. The data
also met the assumption of non-zero variances, with all variance values above
1.
Analysis of demographic data and all psychological variables
Descriptive statistics were generated for participant characteristics and
psychological variables at T1 and T2.
Comparisons with normative data
Mean scores for each measure at T1 (n=90) and T2 (n=69) were calculated. A
series of z-tests for means were conducted comparing the means for the total
sample at T1 (n=90) with normative data for each measure (See Appendix H for
full details). Effect sizes were measured using Cohen’s d [64].
Stepwise regressions
A planned series of stepwise regression analyses were conducted allowing each
predictor to compete for variance in outcome variables. Firstly, interrelationships
between variables at T1 were analysed. Secondly, changes in psychological
variables from T1 to T2 were analysed to identify the variance in each of the
99
outcome variables (health anxiety, quality of life, distress) at T2 accounted for by
mental defeat, existential concerns, beliefs about emotions and intolerance of
uncertainty at T1. Further regressions were conducted to determine the variance
accounted for by time since treatment.
Results
Participant characteristics
The final sample comprised 90 participants, recruited from the Macmillan
Cancer Voices website (88%), Great Western Hospital, Swindon (4.4%),
Salisbury District Hospital (3.2%) and two Breast and Head and Neck cancer
support groups (4.4%). Descriptive analyses of baseline demographic and
treatment-related participant characteristics are summarised in Tables 1 and 2.
Participant characteristics of the total sample were comparable to other UK
samples of cancer patients who had completed treatment with curative intent
[65].
100
Table 1.
Baseline demographic and treatment-related characteristics of the total sample
at T1 and participants that completed questionnaires at T1 and T2.
T1 (n=90) T1 and T2 (n = 69)% %
Demographic characteristicsAge (years) Mean (SD) 53.8
(11.1)53.7 (10.8)
Median 55 55 Range 23–80 27 – 76Gender Male 26 28.9 20 29.0 Female 64 71.1 49 71.0Medical characteristicsCancer site Breast 36 40.0 28 40.6 Gastrointestinal 10 11.1 8 11.6 Lung 1 1.1 1 1.4 Haematological 4 4.4 3 4.3 Gynaecological or Genitourinary
23 25.6 17 24.6
Bone or skin 7 7.8 5 7.2 Head and neck 9 10.0 7 10.1Type of primary treatment Surgery 65 72.2 52 75.4 Radiotherapy 8 8.9 3 4.3 Chemotherapy 17 18.9 14 20.3Number of treatment types One 19 21.1 13 18.8 Two 37 41.1 28 40.6 Three 20 22.2 14 20.3 Four 14 15.6 14 20.3Time since treatment (years) Mean (SD) 2.9 (2.5) 2.8 (2.2) Median 2 2 Range 0.1 – 11.4 0.1 – 10.0Other health conditions None 25 28.1 22 31.9 One or more 64 71.9 47 68.1Table 2.
101
Baseline demographic and treatment-related characteristics of participants at T1
that completed questionnaires at T1 only (n = 21).
%Demographic characteristicsAge (years) Mean (SD) 54.2 (12.2) Median 54.5 Range 23 – 80Gender Male 6 28.6 Female 15 71.4Medical characteristicsCancer site Breast 8 38.1 Gastrointestinal 2 9.5 Lung 0 0 Haematological 1 4.8 Gynaecological or Genitourinary
6 28.6
Bone or skin 2 9.5 Head and neck 2 9.5Type of primary treatment Surgery 13 61.9 Radiotherapy 5 23.8 Chemotherapy 3 14.3Number of treatment types One 6 28.6 Two 9 42.9 Three 6 28.6 Four 0 0Time since treatment (years) Mean (SD) 3.2 (3.5) Median 2.1 Range 0.1 – 11.4Other health conditions None 3 15 One or more 17 85
102
Levels of mental defeat, existential concerns, beliefs about emotions,
intolerance of uncertainty, health anxiety, quality of life and distress in cancer
survivors
Means and standard deviations for the total sample for each predictor variable at
both time points are summarised in Table 3. There was not an overall decrease
in outcomes from T1 to T2, which has previously been observed in prospective
research [e.g. 66]. A series of Z tests for means were conducted to compare
means for the current sample with normative data (See Appendix H for full
details). Results revealed that for mental defeat, the sample was comparable to
community volunteers with chronic pain [16]. The sample scored significantly
lower than primary care outpatients for existential concerns [54]. For IOU, the
current sample scored lower than patients with Generalised Anxiety Disorder
and higher than controls [67]. For health anxiety, 52.2% of the sample scored 15
or over, indicating high health anxiety, with a further 38.9% scoring 18, denoting
people who are likely to meet criteria for a diagnosable disorder. The mean was
also just above 15. The current sample showed significantly lower levels of
health anxiety compared to people with a diagnosis of hypochondriasis, but
were comparable to people with anxiety [57]. The sample scored significantly
lower than breast cancer patients [61] for QoL, indicating comparatively lower
QoL. Compared to normative data for the general population [68] the current
sample scored significantly higher on the measure of distress.
103
Table 3.
Means and standard deviations for each variable for the total sample,
participants who completed both time points and those that completed T1 only.
Total sample(n = 90)
Completed T1 and T2
(n = 69)
Completed T1 only(n = 21)
Mean (SD) (range) Mean (SD) (range) Mean (SD) (range)
T1
Mental Defeat 14.5 (15.8) (0-66) 13.7 (15.0) (0-64) 16.9 (18.4) (0-66)
Existential concerns 13.6 (6.2) (6-30) 14.1 (6.4) (6-30) 12.0 (5.1) (6-22)
Unhelpful beliefs about emotions
35.0 (15.6) (0-68) 32.8 (14.4) (0-61) 42.3 (17.1) (12-68)
Intolerance of uncertainty
29.8 (8.7) (14 – 55) 29.0 (8.6) (14-55) 32.6 (8.6) (16-52)
Health anxiety 15.3 (6.7) (0-35) 15.1 (6.6) (0-35) 15.9 (7.2) (0-28)
Quality of life 21.9 (4.1) (13..5-29.0) 21.9 (4.2) (13.7-29.0) 22.1 (3.8) (13.5 –
27.5)
Psychological
distress
12.6 (9.6) (0-40) 11.6 (9.3) (0-40) 15.8 (10.0) (0-34)
T2
Mental Defeat - 12.3 (15.4) (0-68)
Existential concerns - 14.2 (6.3) (6-30)
Unhelpful beliefs about emotions
- 31.7 (14.9) (0-72)
Intolerance of uncertainty
- 27.8 (8.7) (16-60)
Health anxiety - 14.3 (6.9) (0-31)
Quality of life - 22.6 (4.0) (13.4-29.1)
Psychological
distress
- 10.9 (9.6) (0-37)
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Relationships between psychological variables
Table 4 summarises the results of cross-sectional stepwise regression analyses
to determine relationships between variables at T1.
Table 4.
Outcome variable
Predictors entered Adjusted R²
R²∆ Std error Std β p
Health anxiety
Intolerance of uncertainty
0.20 0.21 0.07 0.46 0.0001
Burden Intolerance of uncertainty
Intolerance of uncertainty and mental defeat
0.21
0.26
0.22
0.06
0.02
0.030.02
0.47
0.300.29
0.0001
0.0100.011
Avoidance Beliefs about emotions
0.07 0.08 0.05 0.28 0.007
Reassurance seeking
Mental defeat 0.05 0.06 0.06 0.25 0.017
Quality of life Mental defeat
Mental defeat andbeliefs about emotions
0.42
0.46
0.43
0.04
0.02
0.020.02
-0.65
-0.56-0.23
0.0001
0.00010.009
Distress Mental defeat
Mental defeat andIntolerance of uncertainty
0.58
0.61
0.59
0.03
0.05
0.050.09
0.77
0.640.22
0.0001
0.00010.008
Summary of stepwise regression analyses: predictors selected into the models
for health anxiety, quality of life and distress at T1.
∆ = increase
105
Psychological variables associated with health anxiety, poor quality of life and
distress at T1
Intolerance of uncertainty was significantly associated with health anxiety
(Adjusted R² = 0.20). In terms of mores specific health anxiety variables, there
were significant associations between IOU, mental defeat and health anxiety
burden. Together, these factors accounted for 26% of the variance in health
anxiety burden. Unhelpful beliefs about emotions was significantly associated
with avoidance behaviour (Adjusted R² = 0.07), and mental defeat was
significantly associated with reassurance seeking (Adjusted R² = 0.05). Mental
defeat and beliefs about emotions were significantly associated with QoL.
Together, these factors accounted for 46% of the variance in QoL. Mental defeat
and intolerance uncertainty were significantly associated with psychological
distress. Together, these factors accounted for 58% of the variance in distress.
Predictors of change in psychological outcomes between T1 and T2
The results of prospective stepwise regression analyses (absolute level) to
identify predictors of the variance in each of the outcome variables at T2 are
reported in Table 5. Mental defeat at T1 was found to be the strongest predictor
of QoL at T2 accounting for 42% of the variance. Mental defeat was also the
strongest predictor of distress at T2, accounting for 33% of the variance. Health
anxiety, (Adjusted R² = 0.09), avoidance behaviour (Adjusted R² = 0.09) and
reassurance seeking (Adjusted R² = 0.07) at T2 were also predicted by mental
defeat at T1. Intolerance of uncertainty emerged as the strongest predictor of
health anxiety burden, which represents anticipated burden of feared illness(es)
(Adjusted R² = 0.21). Unhelpful beliefs about emotions as measured at T1 was a
significant predictor of change in QoL, accounting for 10% of the variance in
QoL at T2 (R²= 0.10, β= -0.34, p=0.004).
106
Table 5.
Summary of stepwise regression analyses: predictors at T1 selected into the
models for health anxiety, quality of life and distress at T2.
Outcome variable
Predictors entered Adjusted R²
R²∆ Std error Std β p
Health anxiety (T2)
Mental defeat (T1) 0.09 0.10 0.05 0.32 0.007
Burden (T2) Intolerance of uncertainty (T1)
0.21 0.22 0.03 0.47 0.0001
Avoidance (T2)
Mental defeat (T1) 0.09 0.10 0.08 0.32 0.007
Reassurance seeking (T2)
Mental defeat (T1) 0.07 0.08 0.08 0.28 0.020
Quality of life (T2)
Mental defeat (T1) 0.42 0.43 0.03 -0.66 0.0001
Distress (T2) Mental defeat (T1) 0.33 0.34 0.06 0.58 0.0001
∆ = increase
Further analyses
There was a significant negative relationship between time since treatment as
measured at T1 and avoidance related to health anxiety at T1 (R²=0.05, β= -
0.23, p= 0.03, Std error=0.34), demonstrating that avoidance decreased the
longer the time since treatment. Time since treatment was also significantly
associated with quality of life at T1 (R²=0.06, β=0.27, p=0.01, Std error=0.17),
and distress at T1 (R²=0.05, β= -0.24, p=0.03, Std error=0.41). This means that
quality of life improved and distress decreased the longer the time since
treatment. No significant relationships were observed for other variables.
107
Discussion
Health anxiety was, as expected, high at 52.2%, as were levels of mental defeat,
intolerance of uncertainty and psychological distress. Low levels of existential
concerns were found, contrary to expectations. It was also found that intolerance
of uncertainty was related to elevated health anxiety, levels of mental defeat and
beliefs about emotions were related to quality of life and mental defeat and
intolerance of uncertainty were associated with levels of distress. Perhaps most
importantly, changes in health anxiety, quality of life and distress were all
predicted by levels of mental defeat at the first point of measurement.
Levels of health anxiety found in this study are higher than prevalence rates
reported in a previous study of cancer patients (23.4%) [14]. This disparity may
reflect sample differences as Jones, Hadjistavropoulos and Gullickson [14]
included people who had been diagnosed within the past 5 years, and did not
specify their disease status. Health anxiety appears to be a specific and
persistent problem for a large proportion of cancer survivors, over and above
general distress warranting specific intervention.
Mental defeat may be an important mediator of both levels of and variations in
QoL and psychological distress in this population, as it was almost the only
determinant of levels and variations in outcome. This is line with previous
studies in pain patients [16]. This suggests that cancer survivors are
experiencing a deeper impact of cancer eroding their sense of self, rather than
simply negative thinking or low mood. This suggests that this complex construct
which concerns catastrophizing, identity and social role would be an appropriate
focus of treatment.
Intolerance of uncertainty (IOU) was significantly associated with health anxiety,
health anxiety burden and distress at T1. This is consistent with current literature
demonstrating higher levels of IOU were related to higher distress in cancer
patients up to 5 years post-diagnosis [15, 43], and IOU as a moderator between
catastrophic health misappraisals and health anxiety in people with high levels
108
of health anxiety [47]. It is hypothesised that IOU is a risk factor for elevated
health anxiety, particularly health anxiety burden, which represents the
anticipated burden of feared illness(es).
Existential concerns were not found to predict any outcomes in this study. This
is at odds with previous research [17], but may be due to sample differences, as
cancer survivors have not been studied alone aside from in qualitative studies
[44]. Time since treatment was not significantly associated with health anxiety at
T1. This is consistent with previous studies [12-14], suggesting that health
anxiety is a persistent problem for cancer survivors.
Clinical implications
This study demonstrates that elevated health anxiety was present in cancer
survivors, up to 11 years post treatment. The transition from the treatment phase
of cancer to remission can be a difficult time for cancer survivors as contact with
clinicians decreases in frequency and the routine and safety of the hospital
system is less prominent [69]. It is likely that high levels of health anxiety are not
currently being recognised or dealt with sufficiently well by healthcare services
and therefore represents an unmet need. This may be because it is being
thought of as anxiety and depression as they present in mental health settings,
and not in the nuanced way proposed by this study. Tyrer et al [70] suggests
that significant health anxiety may be under recognised as it can be
overshadowed by existing medical conditions and other mental health
difficulties. Furthermore, health anxiety is likely to persist in the absence of
treatment [69] and therefore may cause prolonged distress and burden on
healthcare services [69]. Detecting and managing elevated health anxiety in
cancer survivors and ensuring patients receive psychosocial support is therefore
essential.
Thewes et al [71] conducted a qualitative study of medical, nursing and
psychosocial professionals’ perceptions and management of FCR. FCR was
109
perceived as common and challenging to manage, with 99% of participants
reporting a training need in this area. The high levels of health anxiety found in
this study point to the importance of specific training in the psychosocial
management of health anxiety. Health anxiety and FCR can be managed in
similar ways according to the respective cognitive behavioural models.
The Manual for Cancer Services: Psychological Support Measures [72] states
that the provision of psychosocial care by health care professionals at Level 2
should include anxiety management. At Levels 1 and 2 it should include general
psychological support. The lack of knowledge in managing behaviour associated
with elevated health anxiety, such as reassurance seeking may reflect the fact
that level 2 training includes general anxiety management, but does not
delineate how to recognise and manage specific difficulties such as health
anxiety. It would be beneficial to include health anxiety in the level 2 training. It is
important that such training covers the nuances in how people might present, for
example in terms of avoidance and/or reassurance seeking, and how people
can respond consistently among the team.
Due to increasing survivorship and restraints on resources, follow up care for
cancer survivors is currently undergoing service redesign, moving from routine
face to face appointments to supported self-management [73]. This comprises a
tailored approach based on patients self-managing their follow up by triggering
their return for advice and guidance. Patients are prepared for this approach
with workshops which teach skills such as effective self-monitoring. It would be
beneficial at this point to include psycho-education about health anxiety, and
emphasise the importance of appropriate levels of self-monitoring, and seeking
reassurance from medical sources. Self-management may pose difficulties for
those patients with elevated health anxiety that may seek excessive
reassurance, as contact with the team decreases. Patients who might be
avoidant of healthcare systems due to high health anxiety may seek little follow
up care, or miss signs of cancer recurrence or other health problems. Further
110
research is needed to consider the management of health anxiety and distress
in relation to this new service context.
Levels of mental defeat were elevated in this sample, and were an important
predictor of levels of and change in QoL, distress and health anxiety. It would be
important for clinicians to be aware of this issue, in that although treatment has
been successful, many people will feel ‘defeated’ by their experience of cancer,
which represents a risk factor for poorer psychological outcomes. Educating
clinicians about this more complex response and how it might differ from
depression is important in ensuring patients receive targeted psychosocial
support.
Research implications
The finding that health anxiety is elevated in a large proportion of cancer
survivors warrants further research in this area. The cognitive model of health
anxiety posits that health anxiety increases when people feel more vulnerable,
perceive the medical condition to be distressing, feel they are unable to cope
with it and believe that their resources to cope with it are inadequate. Further
examination of how these variables contribute to health anxiety in cancer
survivors is warranted.
Mental defeat and intolerance of uncertainty were significantly associated with
health anxiety, distress and quality of life in this study. As this construct is a
relatively new construct implicated in distress and disability in chronic pain, it is
pertinent to investigate the relationship between these variables in this
population in order to develop empirically derived treatment targets. This study
used a heterogenous sample of cancer survivors, which has offered evidence
that high levels of health anxiety and mental defeat are present. However,
detailed investigation of the constructs studied here are needed within more
homogenous groups based on cancer site, to ensure that the unique
phenomenology of different cancer types, sites and treatments are taken into
111
account in considering psychological outcomes. This is important, as different
cancer types have varying levels of risk of recurrence and invasiveness of
treatment. Therefore, it would also be of value to compare the variables
considered in this study across tumour sites. Furthermore, as we did not place a
time limit on time since treatment, participants were at varying stages of
survivorship. It would be important in future research to consider how
psychological outcomes change according to time since diagnosis and/or
treatment.
Further research is needed to elucidate factors involved in the development of
health anxiety in cancer survivors. The cognitive behavioural model of health
anxiety posits that health anxiety may arise from personal experience of a
distressing illness or treatment, or that of a relative or friend, which may include
misdiagnosis or medical mismanagement [31]. Future research in investigating
differences in health anxiety according to experiences of diagnosis and
treatment of cancer, and past history of illness is warranted. As the first study
investigating health anxiety in cancer survivors, we do not know whether
participants were responding to the health anxiety questionnaire primarily in
relation to cancer as a serious illness (in terms of FCR), or other illnesses.
Health anxiety may concern one or many illnesses, but previous studies have
not made reference to this issue. This also relates in part to whether participants
were anxious about their health prior to having cancer. It would be of interest in
future research to explore this issue further to allow targeted interventions for
FCR or health anxiety to be developed based on the unique phenomenology of
fears in this population.
Intervention research is a priority, for both health anxiety itself in this population
and risk factors such as mental defeat. Although the evidence base for
treatment of clinically significant FCR in cancer survivors is being established
[48, 49], the treatment of health anxiety in this population has not been
considered. CBT has been identified as an effective treatment for health anxiety
in people with medical conditions [33] but this includes a heterogeneous
112
population. Specific research addressing the needs of cancer survivors with
elevated health anxiety needs to take place, taking into account the possibility of
recurrence and need for self-monitoring.
Mental defeat levels were found to be high in the current sample, and was an
important predictor of psychological outcomes. A number of treatment
approaches may be appropriate in reducing levels of mental defeat. Acceptance
and Commitment Therapy (ACT) approaches [74] may be helpful, as they use
acceptance and mindfulness strategies to encourage acceptance of thoughts
and feelings, combined with commitment and behaviour change strategies to
increase psychological flexibility. Furthermore, given the self-critical thinking and
sense of loss of social role and identity that accompanies mental defeat,
compassion-focused approaches [75] may also be helpful, in activating
soothing, affiliative responses to difficult thoughts and feelings such as mental
defeat. Research in these areas may be of value.
Limitations
The current study has methodological strengths in terms of the relatively large
sample size with an achieved power of 0.83. Furthermore, the prospective
design enabled predictors to be identified and there was a satisfactory response
rate from T1 to T2 of 80.23% (excluding those who no longer met criteria).
However, there are limitations which should be considered.
Participants who dropped out of the study following measurement at T1 showed
higher levels of mental defeat, beliefs about emotions, intolerance of uncertainty,
health anxiety, and psychological distress, in addition to poorer QoL. This may
have affected regression results. It would have been valuable to know what
predicted these outcomes at T2 in this group. It is speculated that higher levels
of distress may have been a reason why these participants dropped out,
particularly given more negative beliefs about the experience and expression of
emotions.
113
Seventy-two percent of participants had one or more health conditions. Due to
the high prevalence of health conditions in the sample, and their heterogeneous
nature it was not practicable or meaningful to report proportions or exclude
those with serious health conditions. Some research has shown that the
presence of other health conditions other than cancer may moderate health
anxiety levels [76]. This may have therefore skewed results in terms of elevated
levels of health anxiety related to other health conditions. However, Jones,
Hadjistavropoulos & Gullickson [14] reported that close to half of their sample
had one or more health conditions (47.4%), and presence of other health
problems did not predict health anxiety. Furthermore, there is an increased
incidence of chronic illnesses in long term cancer survivors; attributable to
underlying lifestyle and or treatment effects [77], and it could therefore be
argued that the current sample is representative.
The brief time period between baseline and follow up data collection limits the
conclusions that can be drawn about the predictive power of the variables
studied over longer periods of time. However, this research is exploratory, and
future research should focus on the duration of the post-treatment phase.
Conclusion
This study is the first to explore predictors of health anxiety, quality of life and
distress in UK cancer survivors. It provided an estimate of the extent to which
health anxiety affects cancer survivors, demonstrating that clinically elevated
health anxiety is high in this population, representing an under recognised
problem. It contributes to the literature by demonstrating that mental defeat
predicted changes in health anxiety, quality of life and distress. These findings
highlight the importance of the detection and consideration of the risk factors
underlying elevated health anxiety, psychological distress and poor quality of life
which may be an appropriate target for treatment. As an exploratory study, this
research has also identified health anxiety in cancer survivors as a new and
114
important area for future research, particularly given the rising population of
cancer survivors and increasing focus on self-management in the post-treatment
phase.
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Executive summary: Main research project
Predictors of change in health anxiety, quality of life and psychological distress in UK cancer patients in remission: A prospective study.
This report provides a summary of the findings of a study of factors that predict
change in levels of health anxiety, quality of life and psychological distress in a
sample of UK cancer survivors. The term ‘cancer survivors’ refers to people who
have been diagnosed with and treated for cancer, and are thought to be cured of
the disease or 'in remission'. There are a growing number of cancer survivors in
the UK, largely due to medical advances in the detection and treatment of
cancer, and an ageing population.
The transition from the treatment phase of cancer to remission can be a difficult
time for cancer survivors, both physically and psychologically. A number of
unmet needs have been identified in cancer survivors. Evidence suggests that
though most people adjust well following treatment with curative intent, a
significant proportion develop psychological difficulties, leading to poorer quality
of life. Indeed, research into the experiences of cancer survivors has shown that
these psychological difficulties are more of a priority than physical problems.
This is reflected in the recent focus by the UK government on improving
psychological care for this population (National Cancer Survivorship Initiative,
2010).
Typically, researchers have approached psychological adjustment in cancer
survivorship by determining rates and of broad psychiatric diagnoses such as
anxiety and depression, derived in mental health populations, and factors
associated with these. Anxiety in particular has been shown to be elevated in
cancer survivors. However, applying such diagnoses in the context of cancer
survivorship may not be a particularly helpful or clinically meaningful way to
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understand distress as a reaction to potentially life-threatening illness. A more
nuanced psychological approach is warranted in order to understand the ways in
which psychological variables mediate the impact of serious physical illness. By
implication, this could suggest possible general and more targeted interventions.
Psychological factors likely to interact with medical conditions include those
connected with anxiety. Fear of Cancer Recurrence (FCR) is common in cancer
survivors and is related to health anxiety, which is intense and persistent fear
that one has a serious illness. Health anxiety has been observed in other
medical and non-medical populations including cancer patients. Both constructs
exist on a continuum, from minimal to significant and excessive. Intolerance of
uncertainty (IOU) is a construct that relates to how a person perceives,
interprets, and responds to uncertain situations. It has been well researched in
the development and maintenance of anxiety, and more recently in relation to
health anxiety in cancer survivors.
Linked to but probably separate from low mood, Mental Defeat is a construct
similar to depression as commonly experienced by cancer survivors, which has
been shown to predict higher levels of anxiety and low mood in patients with
pain. Existential concerns relating to issues such as anxiety about death and
lacking meaning in life have also been noted in cancer survivors and have been
found to be associated with lower quality of life and wellbeing. Unhelpful beliefs
about the experience and expression of emotions in general have been
implicated in the maintenance of distress in a number of conditions, including
physical health problems such as chronic fatigue syndrome.
This study investigated whether mental defeat, existential concerns, beliefs
about emotions and intolerance of uncertainty predict levels of health anxiety,
quality of life, depression and anxiety in cancer patients in remission. A
prospective questionnaire design was used. Ninety participants aged 23-80, who
had completed cancer treatment with curative intent were recruited from two
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hospitals, support groups and the Macmillan website. Self-report questionnaires
were used to measure mental defeat, existential concerns, beliefs about
emotions and intolerance of uncertainty, health anxiety, quality of life, depression
and anxiety at two time points, 4 weeks apart.
High levels of health anxiety were reported in 52.2% of the sample. Low levels
of existential concerns were found, contrary to expectations. It was also found
that intolerance of uncertainty was related to elevated health anxiety, levels of
mental defeat and beliefs about emotions were related to quality of life and
mental defeat and intolerance of uncertainty were associated with levels of
distress. Changes in health anxiety, quality of life and distress were all predicted
by levels of mental defeat. This demonstrates that mental defeat and intolerance
of uncertainty are risk factors that could be identified and targeted in treatment.
This study provides evidence that elevated health anxiety is high in cancer
survivors, and highlights the importance of detection and consideration of the
risk factors underlying elevated health anxiety, psychological distress and poor
quality of life, in order to facilitate early detection and treatment. It is likely that
high levels of health anxiety are not currently being recognised or dealt with
sufficiently well by healthcare services and therefore represents an unmet need.
As an exploratory study, this research has also identified health anxiety in
cancer survivors as a new and important area for future research, particularly
given the rising population of cancer survivors and increasing focus on self-
management in the post-treatment phase.
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Connecting narrative
Choice of research areas
I have focused my main project, service improvement project and three case
studies within the area of Clinical Health Psychology, which stemmed from my
interest in this area. I have found that research in both adult and paediatric
clinical health has often been relatively behind that of mental health. This is
reflected in my main project concerning predictors of health anxiety in cancer
survivors, and service improvement project on the psychological and emotional
support needs of parents of children with life-limiting illness, which both
examined needs of these populations in relatively new areas.
I have found that the diversity of health problems and psychological
presentations within clinical health requires a broad range of methodologies. For
example, qualitative methods were most appropriate for exploring the
psychological and emotional support needs of parents with life-limiting or life-
threatening conditions, as they were a hard to reach population with rich and
varied experiences, partly because of the vast nature of conditions of children
within the service. I feel that Clinical Psychology has a great deal to offer in
terms of applied research in physical health settings and is understandably a
growth area.
Similarly in terms of the field of mental health, I have been drawn to areas that
are under-researched, choosing to undertake my literature review in
psychological models of anxiety of ASD in children and adolescents, and case
studies in Cognitive Behavioural Therapy (CBT) for self-esteem in long standing
psychosis, and CBT for generalised anxiety disorder in an individual with a mild
learning disability. I have been particularly enthused by applying new models in
line with research developments in case studies rather than routine clinical work
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with well-established models. I am grateful that the programme has encouraged
these opportunities, and appreciate the importance as a scientist-practitioner in
contributing to the development of research and practice.
Personal reactions
I initially found being the chief investigator in a number of projects daunting, but
was more comfortable with case studies as felt these related directly to my
clinical work and I had completed similar assignments for my Postgraduate
Certificate in Low Intensity Psychological Therapies. Prior to training, I had
limited experience of research, aside from assisting with the write up of a
qualitative project and having small roles in audit projects. I felt the pressure to
come up with a novel, ground-breaking research question and design that I
would enjoy carrying out and ultimately could be completed within the time-
frame. I expected other Trainee Clinical Psychologists in my cohort to have a
project already in mind from pre-training interests and research experience.
However, I found the comprehensive teaching on research methods and
statistics supported my learning and I was introduced to research in a gradual
way. Completing problem based learning presentations and an assignment
involving critical appraisal of quantitative and qualitative research papers were
particularly helpful in combining my knowledge from my undergraduate degree
and the DClinPsy programme.
The research process
I did not have a clear idea of the research areas I was interested in prior to
training. In my first year I attended the research fair that was designed to expose
Trainees to research activity in the region and discuss research ideas with
potential supervisors.
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I found this event energising and inspiring in terms of the research interests and
activity in the region and the commitment to supporting Trainees in developing
their research ideas. Moving through each of the different placements and
having teaching throughout training I have found that I noticed opportunities for
research on many occasions, and if I had the time and scope would have
followed up on them. This experience has taught me that gaps in the literature
and areas for future research and audit are vast, and I will be able to utilise my
broad knowledge base and research skills to identify and act upon these in my
future career.
I met the supervisor for my Service Improvement Project, Jackie MacCallam at
the research fair and began discussing ideas initially in individual meetings with
her. Preliminary ideas focused on identifying the prevalence and predictors of
late onset trauma in parents of children with life-limiting conditions. This was
borne out of my supervisor’s interests and clinical observations. It soon became
apparent that her thinking was that this would lend itself more to a main project
than a service improvement project. I learned that good communication and
discussion of the scope of the project is important in the early stages of research
development in terms of being sure of the expectations and needs of
collaborators. We tried to reduce the scope and focus of the study, whilst
retaining a novel element but remaining useful to the service. I found this
process particularly difficult in terms of being keen to keep the project in line with
my supervisor and the services’ interests. Once the focus of the research had
shifted from prevalence and predictors of a specific presentation, we were able
to think more broadly about the needs of the service in terms of emotional
support needs and barriers to seeking help, and found that the new project fitted
more, in a rich sense with understanding why people with trauma symptoms and
other difficulties might not seek help earlier. Initially we tried to construct a
questionnaire that would capture these elements, but realised a qualitative
design would be more appropriate. On reflection, I am pleased that we changed
the research design to reflect the needs of the service, and improve feasibility. I
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was also pleased to have the opportunity to conduct qualitative research, to
widen the focus of my research portfolio and provide me with skills in this
methodology.
Recruitment was a challenging element of this project. My supervisor was
confident that we would be able to recruit participants easily from this
population, given that the service covered a wide geographical base and similar
research had been carried out before. However, there was a low response rate
to invitations to take part. If I were able to approach this again, I would be sure
to engage with other clinicians in the team to discuss the process of recruitment
for previous studies in terms of what worked well and what did not. I would also
consider other ways of recruiting such as involving clinicians in recruiting and
carrying out the process over a longer period. In a discussion with a Trainee
from another course who conducted their research in a similar population had
similar unanticipated difficulties, reflecting that parents with children with life-
limiting conditions are a ‘hard to reach; population in relation to recruitment for
research. I understand that she had to use an action research approach,
immersing herself in their communities in order to build relationships. As this
was a service related project alongside other research commitments this would
not have been possible. I have learned that it is important to carefully consider
and research the population that you choose to recruit from, and consider how
this has been successfully managed in the past.
In terms of conducting semi-structured interviews, I felt that this required a lot of
skill in relation to ensuring you do not influence participants’ responses. This
seemed an obvious contrast to the skill of questioning in a therapy context. I
found that reminding myself of my ‘researcher’ role in this context was helpful,
and will be mindful of the importance of this awareness in future qualitative
research. On reflection, I feel that I did not have much supervision or any
teaching on the process of conducting interviews; the focus was more on the
construction of interview schedules and analysis. I would be pro-active in future
134
in seeking this out. I found that my field supervisor had relatively less research
experience when compared to the supervisor for my main project, and found
that I had to be more autonomous in deciding on the write up the research.
My main project required NHS and research and development ethical approval.
Ethical approval was a quick and easy process in my experience, but due to
local organisational issues research and development approval at one site took
much longer, significantly delaying the recruitment process. On reflection, this
should have been anticipated earlier. However, the recruitment process has still
been challenging in terms of participants from NHS sites. Although I would have
preferred to obtain a more representative sample through NHS sites, the
majority of participants were recruited from Macmillan cancer voices, where
people can sign up to give their time to projects including research. This proved
invaluable in reaching the number of participants I needed, and came about due
to my own creative thinking about recruitment opportunities. This has taught me
a valuable lesson in terms of being flexible and creative in finding sources of
recruitment, but also to thoroughly consider feasibility of the project. On
reflection, it seems the project was a large undertaking for a doctoral project.
This has been influenced by my main project academic supervisor, who often
suggested designs with a large scope that would be unfeasible within the
timeframe.
I found that the extent to which I exercised autonomy in my main research
project varied among different elements of the project. I was solely responsible
for scoping and investigating the literature and in realising the final design, and
applying for ethical approval. Having not completed an application for ethical
approval before, I found it to be a lengthy process but learned that it can be a
straightforward process if you are as clear as possible in the protocols. I was
more influenced by supervisors in terms of the variables measured and use of
measures. I found this particularly challenging when I had three supervisors at
the project’s initiation, but this felt easier when I had two supervisors as one left
135
the programme. Supervisors wanted to incorporate their own interests into the
study, but I felt this still fitted with my research interests. The main outcome
variable measured was health anxiety, which fitted with my supervisor’s
interests, but on researching the field further and carrying out the study I feel
that measuring fear of recurrence in addition would have been helpful. This idea
came to mind once research and development approval had almost been
obtained, so would have delayed the project further if an amendment was
submitted. However, I was mindful of not focusing on too much, and having to
draw the line somewhere in terms of variables to be included.
In terms of recruitment, due to ethical reasons I could not approach participants
directly. My supervisor put me into contact with clinicians at Great Western
Hospital, Swindon and I had already built relationships with clinicians at
Salisbury District Hospital through my placement there. I produced and delivered
presentations to various teams at Great Western Hospital, with good responses.
The teams were interested in the research and could see the clinical relevance
of studying health anxiety in this population. However, I learned of the
importance of regularly checking with clinicians if they had any suitable patients,
and recruitment was possible through the efforts of a handful of enthusiastic
nurses and Consultants. I found that utilising these relationships to build
momentum in recruitment was more resource efficient than continually trying to
involve more of the team. Similarly, I found recruitment was more successful at
Salisbury District Hospital through the professionals I had already had contact
with in my clinical work. Being present in the hospital I think gave more of a
sense of the project being directly beneficial to their practice and patients,
increasing their motivation to recruit patients to the study. Keeping up with a log
of participants and corresponding with them required a great deal of
administration resource. I managed this by working during the evenings after
placement, but learned this was essential to maintain the momentum of the
project. In future, I would consider the use of resources of Assistant
Psychologists or placement students to assist with this, but nonetheless it has
136
provided me with insights into the challenges of fitting in research around
placements with limited study time.
I see the value and importance of involving service users in the development
and realisation of research. In terms of my main research project, one person
with personal experience of cancer completed the questionnaires to assess time
taken to complete the questionnaires and acceptability. I would have liked to
incorporate service user involvement on a larger scale, and considered using
the Thames Valley Cancer Research Network to gain feedback at an earlier
stage and pilot questionnaires, but time constraints made this unfeasible. In my
future research work, I would like to ensure this is considered more carefully at
the planning stages to ensure it is not overlooked.
In terms of data analysis, I found that practical support of carrying out the
analyses under supervision was beneficial to my learning, particularly in dealing
with challenges of applied research such as missing data. I now feel I could
approach multivariate analyses, often used in Clinical Psychology with this
grounding in practical knowledge of analysis and how this should be
disseminated.
My research outline, submitted early on in the first year on reflection is very
different to the final projects I carried out, and I perhaps did not expect this
evolutionary process to be so great. I was frustrated with this at first, but saw
that others in my cohort were going through a similar process. I have now come
to accept that the evolution and often transformation of ideas is a process
inherent in research, and not necessarily a negative one. Coming to understand
that research is not a linear process has been challenging, but exciting. I have
found that the final projects, when compared with the embryonic ideas I set out
with are much closer to the current state of the respective research fields, needs
of the services and clinical context.
137
Future aspirationsThrough training as a Clinical Psychologist I have been able to develop a sound
knowledge of the process of initiating, developing, implementing, analysing,
writing up and disseminating research in ways appropriate to the NHS context. I
have particularly come to be aware of the need for research to be relevant and
useful to the service or population in which it takes place. Additionally, I have
become more aware of the economic and political drivers shaping research and
how Clnical Psychologists can be an asset in demonstrating the value of
psychological approaches in meeting mental health and service needs.
I would like to continue to be actively involved in conducting research either in
collaboration with colleagues or supervising others’ work throughout my career.
When thinking initially about disseminating my work I was not sure it was of a
sufficient standard. However, having submitted and been accepted to present all
of my projects and case studies aside from two at national and international
conferences I have come to realise that the field encourages dissemination of
novel, good quality research from early career researchers. I have also valued
the opportunity to disseminate my work and add to the field in this respect, as
well as networking with colleagues.
I feel that clinical research and audit are fundamental skills as a Clinical
Psychologist, especially given the challenge of retaining and demonstrating the
unique skills of a psychologist compared with other professionals able to provide
therapy. I plan to do this through maintaining links with the programme and
previous supervisors if possible, as well as seeking out local research networks
and opportunities as a qualified psychologist. The post-qualification jobs I have
been interested in have emphasised research and audit as a key element of the
role, so I hope to agree time to devote to research and audit early on in my
career. I realise that this is likely to come with barriers such as pressures arising
from clinical commitments and organisational targets to meet for clinical activity.
138
I am hopeful I can remain aware of these and balance them as far as possible. I
am concerned that research may not be viewed as a priority or a valuable
resource. I hope to demonstrate the utility of research by being pro-active in
identifying areas for service evaluation or improvement, in order to demonstrate
efficacy and cost effectiveness of different aspects of my work and thus highlight
the value of elements of the service to commissioners. I realise that most
qualified psychologists do not remain research active following qualification.
However, being aware of this trend prior to completion of training has allowed
me to reflect on these issues prior to qualifying.
I am aware that resources to complete each stage of the research process will
be limited. If I cannot employ the assistance of colleagues such as placement
students or assistant psychologists I plan to engage in smaller scale activities
such as case studies. During my training, I have found simple A-B designs to be
beneficial in allowing me to consider the context and evidence base for my work,
and lead to improvements in my clinical practice. By continually critically
appraising research and applying it to my work I have been able to develop
theory-practice links that ensure that my work is truly ethical and evidence
based. In addition, single case designs allow for the exploration of novel
problems and interventions, which is an exciting prospect and has much to offer
in terms of theory and treatment development. As a Trainee, I have not been
involved in grant proposals or funding of research projects. One of my goals on
completion of training is to find out about this process through individual
research, liaising with senior colleagues and researchers in the area.
I will also join local special interest groups and CPD opportunities. Developing
links such as these have allowed me to network with clinicians with similar
interests and aspirations. Once qualified, I hope that these will foster
collaborative relationships.
139
Appendices
Appendix A: Instructions to authors: Critical Literature Review……………..…141
Appendix B: Instructions to authors: Service improvement project………...….165
Appendix C: Instructions to authors: Main research project…………..………..171
Appendix D: Interview schedule: Service improvement project………………..184
Appendix E: Questionnaires: Main research project…………………………….187
Appendix F: Description of questionnaire measures: Main research project…207
Appendix G: Ethical approval documentation: Main research project…………211
Appendix H: Z test comparisons with normative data: Main research project..221
140
Appendix A: Instructions to authors: Critical Literature Review
Editorial Procedure
The Journal uses a double-blind review process. Therefore, when submitting a
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Types of papers
Review Articles:
The preferred article length is 20-23 manuscript pages long (not including
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Review your manuscript for these elements
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141
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References
Appendix
Figure Caption Sheet
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144
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The third paragraph is Acknowledgments. It identifies grants or other
145
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Italic for single letters that denote mathematical constants, variables,
and unknown quantities
Roman/upright for numerals, operators, and punctuation, and
commonly defined functions or abbreviations, e.g., cos, det, e or exp,
lim, log, max, min, sin, tan, d (for derivative)
Bold for vectors, tensors, and matrices.
Citation
Cite references in the text by name and year in parentheses. Some examples:
Negotiation research spans many disciplines (Thompson 1990).
This result was later contradicted by Becker and Seligman (1996).
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This effect has been widely studied (Abbott 1991; Barakat et al. 1995;
Kelso and Smith 1998; Medvec et al. 1999).
Reference list
The list of references should only include works that are cited in the text and that
have been published or accepted for publication. Personal communications and
unpublished works should only be mentioned in the text. Do not use footnotes or
endnotes as a substitute for a reference list.
Reference list entries should be alphabetized by the last names of the first
author of each work.
Journal article
Harris, M., Karper, E., Stacks, G., Hoffman, D., DeNiro, R., Cruz, P., et al.
(2001). Writing labs and the Hollywood connection. Journal of Film
Writing, 44(3), 213-245
Article by DOI
Slifka, M. K., & Whitton, J. L. (2000) Clinical implications of dysregulated
cytokine production. Journal of Molecular Medicine,
doi:10.1007/s001090000086
Book
Calfee, R. C., & Valencia, R. R. (1991). APA guide to preparing
manuscripts for journal publication. Washington, DC: American
Psychological Association.
Book chapter
O’Neil, J. M., & Egan, J. (1992). Men’s and women’s gender role
journeys: Metaphor for healing, transition, and transformation. In B. R.
Wainrib (Ed.), Gender issues across the life cycle (pp. 107–123). New
York: Springer.
Online document
Abou-Allaban, Y., Dell, M. L., Greenberg, W., Lomax, J., Peteet, J.,
Torres, M., & Cowell, V. (2006). Religious/spiritual commitments and
psychiatric practice. Resource document. American Psychiatric
147
Association.
http://www.psych.org/edu/other_res/lib_archives/archives/200604.pdf.
Accessed 25 June 2007.
Journal names and book titles should be italicized.
For authors using EndNote, Springer provides an output style that supports the
formatting of in-text citations and reference list.
Tables
All tables are to be numbered using Arabic numerals.
Tables should always be cited in text in consecutive numerical order.
For each table, please supply a table caption (title) explaining the
components of the table.
Identify any previously published material by giving the original source in
the form of a reference at the end of the table caption.
Footnotes to tables should be indicated by superscript lower-case letters
(or asterisks for significance values and other statistical data) and
included beneath the table body.
Each table should be inserted on a separate page at the back of the manuscript
in the order noted above. A call-out for the correct placement of each table
should be included in brackets within the text immediately after the phrase in
which it is first mentioned. Copyright permission footnotes for tables are typed
as a table note.
Artwork and illustration guidelines
For the best quality final product, it is highly recommended that you submit all of
your artwork – photographs, line drawings, etc. – in an electronic format. Your
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art will then be produced to the highest standards with the greatest accuracy to
detail. The published work will directly reflect the quality of the artwork provided.
Electronic Figure Submission
Supply all figures electronically.
Indicate what graphics program was used to create the artwork.
For vector graphics, the preferred format is EPS; for halftones, please use
TIFF format. MSOffice files are also acceptable.
Vector graphics containing fonts must have the fonts embedded in the
files.
Name your figure files with "Fig" and the figure number, e.g., Fig1.eps.
Line Art
Definition: Black and white graphic with no shading.
Do not use faint lines and/or lettering and check that all lines and lettering
within the figures are legible at final size.
All lines should be at least 0.1 mm (0.3 pt) wide.
Scanned line drawings and line drawings in bitmap format should have a
minimum resolution of 1200 dpi.
Vector graphics containing fonts must have the fonts embedded in the
files.
Combination Art Definition: a combination of halftone and line art, e.g., halftones
containing line drawing, extensive lettering, color diagrams, etc.
Combination artwork should have a minimum resolution of 600 dpi.
Color Art
Color art is free of charge for online publication.
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If black and white will be shown in the print version, make sure that the
main information will still be visible. Many colors are not distinguishable
from one another when converted to black and white. A simple way to
check this is to make a xerographic copy to see if the necessary
distinctions between the different colors are still apparent.
If the figures will be printed in black and white, do not refer to color in the
captions.
Color illustrations should be submitted as RGB (8 bits per channel).
Figure Lettering
To add lettering, it is best to use Helvetica or Arial (sans serif fonts).
Keep lettering consistently sized throughout your final-sized artwork,
usually about 2–3 mm (8–12 pt).
Variance of type size within an illustration should be minimal, e.g., do not
use 8-pt type on an axis and 20-pt type for the axis label.
Avoid effects such as shading, outline letters, etc.
Do not include titles or captions within your illustrations.
Figure Numbering
All figures are to be numbered using Arabic numerals.
Figures should always be cited in text in consecutive numerical order.
Figure parts should be denoted by lowercase letters (a, b, c, etc.).
If an appendix appears in your article and it contains one or more figures,
continue the consecutive numbering of the main text. Do not number the
appendix figures,
"A1, A2, A3, etc." Figures in online appendices (Electronic Supplementary
Material) should, however, be numbered separately.
Figure Captions
Each figure should have a concise caption describing accurately what the
figure depicts. Include the captions in the text file of the manuscript, not in
the figure file.150
Figure captions begin with the term Fig. in bold type, followed by the
figure number, also in bold type.
No punctuation is to be included after the number, nor is any punctuation
to be placed at the end of the caption.
Identify all elements found in the figure in the figure caption; and use
boxes, circles, etc., as coordinate points in graphs.
Identify previously published material by giving the original source in the
form of a reference citation at the end of the figure caption.
Figure Placement and Size
When preparing your figures, size figures to fit in the column width.
For most journals the figures should be 39 mm, 84 mm, 129 mm, or 174
mm wide and not higher than 234 mm.
For books and book-sized journals, the figures should be 80 mm or 122
mm wide and not higher than 198 mm.
Permissions
If you include figures that have already been published elsewhere, you must
obtain permission from the copyright owner(s) for both the print and online
format. Please be aware that some publishers do not grant electronic rights for
free and that Springer will not be able to refund any costs that may have
occurred to receive these permissions. In such cases, material from other
sources should be used.
Accessibility
In order to give people of all abilities and disabilities access to the content
of your figures, please make sure that
All figures have descriptive captions (blind users could then use a text-to-
speech software or a text-to-Braille hardware)
Patterns are used instead of or in addition to colors for conveying
information (colorblind users would then be able to distinguish the visual
elements)151
Any figure lettering has a contrast ratio of at least 4.5:1
Figure caption sheet
The figure caption sheet contains a list of only the captions for all figures used.
Center the label "Figure Captions" in uppercase and lowercase letters at the top
of the page. Begin each caption entry flush left, and type the word "Figure",
followed by the appropriate number and a period, all in italics. In the text of the
caption (not italicized), capitalize only the first word and any proper nouns. If the
caption is more than one line, double-space between the lines, and type the
second and subsequent lines flush left. Table notes: Copyright permission
footnotes for figures are typed as part of the figure caption.
Each figure should appear on a separate page. The page where the
figure is found should have the figure number and the word "top"[ie,
Figure 1 top] typed above the figure. Figures or illustrations (photographs,
drawings, diagrams, and charts) are to be numbered in one consecutive
series of arabic numerals. Figures may be embedded in the text of a
Word or Wordperfect document. Electronic artwork submitted on disk may
be in the TIFF, EPS or Powerpoint format (best is 1200 dpi for line and
300 dpi for half-tones and gray-scale art). Color art should be in the
CYMK color space. Assistance will be provided by the system
administrator if you do not have electronic files for figures; originals of
artwork may be sent to the system administrator to be uploaded. *** After
first mention in the body of the manuscript, a call-out for the correct
placement of each figure should be included in brackets on a separate
line within the text.
Electronic Supplementary Material
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Springer accepts electronic multimedia files (animations, movies, audio, etc.)
and other supplementary files to be published online along with an article or a
book chapter. This feature can add dimension to the author's article, as certain
information cannot be printed or is more convenient in electronic form.
Submission
Supply all supplementary material in standard file formats.
Please include in each file the following information: article title, journal
name, author names; affiliation and e-mail address of the corresponding
author.
To accommodate user downloads, please keep in mind that larger-sized
files may require very long download times and that some users may
experience other problems during downloading.
Audio, Video, and Animations
Always use MPEG-1 (.mpg) format.
Text and Presentations
Submit your material in PDF format; .doc or .ppt files are not suitable for
long-term viability.
A collection of figures may also be combined in a PDF file.
Spreadsheets
Spreadsheets should be converted to PDF if no interaction with the data
is intended.
If the readers should be encouraged to make their own calculations,
spreadsheets should be submitted as .xls files (MS Excel).
Specialized Formats
Specialized format such as .pdb (chemical), .wrl (VRML), .nb
(Mathematica notebook), and .tex can also be supplied.
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Collecting Multiple Files
It is possible to collect multiple files in a .zip or .gz file.
Numbering
If supplying any supplementary material, the text must make specific
mention of the material as a citation, similar to that of figures and tables.
Refer to the supplementary files as “Online Resource”, e.g., "... as shown
in the animation (Online Resource 3)", “... additional data are given in
Online Resource 4”.
Name the files consecutively, e.g. “ESM_3.mpg”, “ESM_4.pdf”.
Captions
For each supplementary material, please supply a concise caption
describing the content of the file.
Processing of supplementary files
Electronic supplementary material will be published as received from the
author without any conversion, editing, or reformatting.
Accessibility
o The manuscript contains a descriptive caption for each supplementary
material
o Video files do not contain anything that flashes more than three times
per second (so that users prone to seizures caused by such effects are
not put at risk)
After acceptance
Upon acceptance of your article you will receive a link to the special Author
Query Application at Springer’s web page where you can sign the Copyright
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Transfer Statement online and indicate whether you wish to order OpenChoice,
offprints, or printing of figures in color.
Once the Author Query Application has been completed, your article will be
processed and you will receive the proofs.
Open Choice
In addition to the normal publication process (whereby an article is submitted to
the journal and access to that article is granted to customers who have
purchased a subscription), Springer provides an alternative publishing option:
Springer Open Choice. A Springer Open Choice article receives all the benefits
of a regular subscription-based article, but in addition is made available publicly
through Springer’s online platform SpringerLink.
Copyright transfer
Authors will be asked to transfer copyright of the article to the Publisher (or grant
the Publisher exclusive publication and dissemination rights). This will ensure
the widest possible protection and dissemination of information under copyright
laws.
Open Choice articles do not require transfer of copyright as the copyright
remains with the author. In opting for open access, the author(s) agree to
publish the article under the Creative Commons Attribution License.
Offprints
Offprints can be ordered by the corresponding author.
Color illustrations
Online publication of color illustrations is free of charge. For color in the print
version, authors will be expected to make a contribution towards the extra costs.
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Proof reading
The purpose of the proof is to check for typesetting or conversion errors and the
completeness and accuracy of the text, tables and figures. Substantial changes
in content, e.g., new results, corrected values, title and authorship, are not
allowed without the approval of the Editor.
After online publication, further changes can only be made in the form of an
Erratum, which will be hyperlinked to the article.
Online First
The article will be published online after receipt of the corrected proofs. This is
the official first publication citable with the DOI. After release of the printed
version, the paper can also be cited by issue and page numbers.
Ethical Responsibilities of Authors
This journal is committed to upholding the integrity of the scientific record. As a
member of the Committee on Publication Ethics (COPE) the journal will follow
the COPE guidelines on how to deal with potential acts of misconduct.
Authors should refrain from misrepresenting research results which could
damage the trust in the journal, the professionalism of scientific authorship, and
ultimately the entire scientific endeavour. Maintaining integrity of the research
and its presentation can be achieved by following the rules of good scientific
practice, which include:
The manuscript has not been submitted to more than one journal for
simultaneous consideration.
The manuscript has not been published previously (partly or in full),
unless the new work concerns an expansion of previous work (please
provide transparency on the re-use of material to avoid the hint of text-
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recycling (“self-plagiarism”)).
A single study is not split up into several parts to increase the quantity of
submissions and submitted to various journals or to one journal over time
(e.g. “salami-publishing”).
No data have been fabricated or manipulated (including images) to
support your conclusions
No data, text, or theories by others are presented as if they were the
author’s own (“plagiarism”). Proper acknowledgements to other works
must be given (this includes material that is closely copied (near
verbatim), summarized and/or paraphrased), quotation marks are used
for verbatim copying of material, and permissions are secured for material
that is copyrighted.
Important note: the journal may use software to screen for plagiarism.
Consent to submit has been received explicitly from all co-authors, as
well as from the responsible authorities - tacitly or explicitly - at the
institute/organization where the work has been carried out, before the
work is submitted.
Authors whose names appear on the submission have contributed
sufficiently to the scientific work and therefore share collective
responsibility and accountability for the results.
In addition:
Changes of authorship or in the order of authors are not accepted after
acceptance of a manuscript.
Requesting to add or delete authors at revision stage, proof stage, or
after publication is a serious matter and may be considered when
justifiably warranted. Justification for changes in authorship must be
compelling and may be considered only after receipt of written approval
from all authors and a convincing, detailed explanation about the
role/deletion of the new/deleted author. In case of changes at revision
stage, a letter must accompany the revised manuscript. In case of
changes after acceptance or publication, the request and documentation 157
must be sent via the Publisher to the Editor-in-Chief. In all cases, further
documentation may be required to support your request. The decision on
accepting the change rests with the Editor-in-Chief of the journal and may
be turned down. Therefore authors are strongly advised to ensure the
correct author group, corresponding author, and order of authors at
submission.
Upon request authors should be prepared to send relevant
documentation or data in order to verify the validity of the results. This
could be in the form of raw data, samples, records, etc.
If there is a suspicion of misconduct, the journal will carry out an investigation
following the COPE guidelines. If, after investigation, the allegation seems to
raise valid concerns, the accused author will be contacted and given an
opportunity to address the issue. If misconduct has been established beyond
reasonable doubt, this may result in the Editor-in-Chief’s implementation of the
following measures, including, but not limited to:
If the article is still under consideration, it may be rejected and returned to
the author.
If the article has already been published online, depending on the nature
and severity of the infraction, either an erratum will be placed with the
article or in severe cases complete retraction of the article will occur. The
reason must be given in the published erratum or retraction note.
The author’s institution may be informed.
Compliance with Ethical Standards
To ensure objectivity and transparency in research and to ensure that accepted
principles of ethical and professional conduct have been followed, authors
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should include information regarding sources of funding, potential conflicts of
interest (financial or non-financial), informed consent if the research involved
human participants, and a statement on welfare of animals if the research
involved animals.
Authors should include the following statements (if applicable) in a separate
section entitled “Compliance with Ethical Standards” before the References
when submitting a paper:
Disclosure of potential conflicts of interest
Research involving Human Participants and/or Animals
Informed consent
Please note that standards could vary slightly per journal dependent on their
peer review policies (i.e. double blind peer review) as well as per journal subject
discipline. Before submitting your article check the Instructions for Authors
carefully.
The corresponding author should be prepared to collect documentation of
compliance with ethical standards and send if requested during peer review or
after publication.
The Editors reserve the right to reject manuscripts that do not comply with the
above-mentioned guidelines. The author will be held responsible for false
statements or failure to fulfill the above-mentioned guidelines.
Disclosure of potential conflicts of interest
Authors must disclose all relationships or interests that could influence or bias
the work. Although an author may not feel there are conflicts, disclosure of
159
relationships and interests affords a more transparent process, leading to an
accurate and objective assessment of the work. Awareness of real or perceived
conflicts of interests is a perspective to which the readers are entitled and is not
meant to imply that a financial relationship with an organization that sponsored
the research or compensation for consultancy work is inappropriate. Examples
of potential conflicts of interests that are directly or indirectly related to the
research may include but are not limited to the following:
Research grants from funding agencies (please give the research funder
and the grant number)
Honoraria for speaking at symposia
Financial support for attending symposia
Financial support for educational programs
Employment or consultation
Support from a project sponsor
Position on advisory board or board of directors or other type of
management relationships
Multiple affiliations
Financial relationships, for example equity ownership or investment
interest
Intellectual property rights (e.g. patents, copyrights and royalties from
such rights)
Holdings of spouse and/or children that may have financial interest in the
work
In addition, interests that go beyond financial interests and compensation (non-
financial interests) that may be important to readers should be disclosed. These
may include but are not limited to personal relationships or competing interests
directly or indirectly tied to this research, or professional interests or personal
beliefs that may influence your research.
The corresponding author collects the conflict of interest disclosure forms from
160
all authors. In author collaborations where formal agreements for representation
allow it, it is sufficient for the corresponding author to sign the disclosure form on
behalf of all authors.
The corresponding author will include a summary statement on the title page that is separate from their manuscript, that reflects what is recorded in the
potential conflict of interest disclosure form(s).
See below examples of disclosures:
Funding: This study was funded by X (grant number X).
Conflict of Interest: Author A has received research grants from Company A.
Author B has received a speaker honorarium from Company X and owns stock
in Company Y. Author C is a member of committee Z.
If no conflict exists, the authors should state:
Conflict of Interest: The authors declare that they have no conflict of interest.
Research involving human participants and/or animals
1) Statement of human rightsWhen reporting studies that involve human participants, authors should include
a statement that the studies have been approved by the appropriate institutional
and/or national research ethics committee and have been performed in
accordance with the ethical standards as laid down in the 1964 Declaration of
Helsinki and its later amendments or comparable ethical standards.
If doubt exists whether the research was conducted in accordance with the 1964
Helsinki Declaration or comparable standards, the authors must explain the
161
reasons for their approach, and demonstrate that the independent ethics
committee or institutional review board explicitly approved the doubtful aspects
of the study.
The following statements should be included in the text before the References
section:
Ethical approval: “All procedures performed in studies involving human
participants were in accordance with the ethical standards of the institutional
and/or national research committee and with the 1964 Helsinki declaration and
its later amendments or comparable ethical standards.”
For retrospective studies, please add the following sentence:
“For this type of study formal consent is not required.”
2) Statement on the welfare of animals
The welfare of animals used for research must be respected. When reporting
experiments on animals, authors should indicate whether the international,
national, and/or institutional guidelines for the care and use of animals have
been followed, and that the studies have been approved by a research ethics
committee at the institution or practice at which the studies were conducted
(where such a committee exists).
For studies with animals, the following statement should be included in the text
before the References section:
Ethical approval: “All applicable international, national, and/or institutional
guidelines for the care and use of animals were followed.”
If applicable (where such a committee exists): “All procedures performed in
studies involving animals were in accordance with the ethical standards of the
institution or practice at which the studies were conducted.”
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If articles do not contain studies with human participants or animals by any of
the authors, please select one of the following statements:
“This article does not contain any studies with human participants performed by
any of the authors.”
“This article does not contain any studies with animals performed by any of the
authors.”
“This article does not contain any studies with human participants or animals
performed by any of the authors.”
Informed consent
All individuals have individual rights that are not to be infringed. Individual
participants in studies have, for example, the right to decide what happens to the
(identifiable) personal data gathered, to what they have said during a study or an
interview, as well as to any photograph that was taken. Hence it is important that
all participants gave their informed consent in writing prior to inclusion in the
study. Identifying details (names, dates of birth, identity numbers and other
information) of the participants that were studied should not be published in
written descriptions, photographs, and genetic profiles unless the information is
essential for scientific purposes and the participant (or parent or guardian if the
participant is incapable) gave written informed consent for publication. Complete
anonymity is difficult to achieve in some cases, and informed consent should be
obtained if there is any doubt. For example, masking the eye region in
photographs of participants is inadequate protection of anonymity. If identifying
characteristics are altered to protect anonymity, such as in genetic profiles,
authors should provide assurance that alterations do not distort scientific
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meaning.
The following statement should be included:
Informed consent: “Informed consent was obtained from all individual
participants included in the study.”
If identifying information about participants is available in the article, the
following statement should be included:
“Additional informed consent was obtained from all individual participants for
whom identifying information is included in this article.”
Appendix B: Instructions to authors: Service improvement project
Prior to submission, please carefully read and follow the submission guidelines
164
detailed below. Manuscripts that do not conform to the submission guidelines
may be returned without review.
Submission
Submit manuscripts electronically (.rtf or .doc) through the Manuscript
Submission Portal.
Jennifer Shroff Pendley
Nemours/Alfred I. duPont Hospital for Children, Wilmington, DE
W. Douglas Tynan
Nemours Health and Prevention Services, Newark, DE
Manuscript Preparation
Prepare manuscripts according to the Publication Manual of the American
Psychological Association (6 th edition) . Manuscripts may be copyedited for bias-
free language (see Chapter 3 of the Publication Manual).
Review APA's Checklist for Manuscript Submission before submitting your
article.
Length and Formatting of Manuscripts
Full-length manuscripts should not exceed 25 pages total (including cover page,
abstract, text, references, tables, and figures), with margins of at least 1 inch on
all sides and a standard font (e.g., Times New Roman) of 12 points (no smaller).
Brief reports and case reports should not exceed 12 pages.
Double-space all copy. Other formatting instructions, as well as instructions on
preparing tables, figures, references, metrics, and abstracts, appear in the
Manual.
Below are additional instructions regarding the preparation of display equations,
computer code, and tables.
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Display Equations
We strongly encourage you to use MathType (third-party software) or Equation
Editor 3.0 (built into pre-2007 versions of Word) to construct your equations,
rather than the equation support that is built into Word 2007 and Word 2010.
Equations composed with the built-in Word 2007/Word 2010 equation support
are converted to low-resolution graphics when they enter the production process
and must be rekeyed by the typesetter, which may introduce errors.
To construct your equations with MathType or Equation Editor 3.0:
Go to the Text section of the Insert tab and select Object.
Select MathType or Equation Editor 3.0 in the drop-down menu.
If you have an equation that has already been produced using Microsoft Word
2007 or 2010 and you have access to the full version of MathType 6.5 or later,
you can convert this equation to MathType by clicking on MathType Insert
Equation. Copy the equation from Microsoft Word and paste it into the MathType
box. Verify that your equation is correct, click File, and then click Update. Your
equation has now been inserted into your Word file as a MathType Equation.
Use Equation Editor 3.0 or MathType only for equations or for formulas that
cannot be produced as Word text using the Times or Symbol font.
Computer Code
Because altering computer code in any way (e.g., indents, line spacing, line
breaks, page breaks) during the typesetting process could alter its meaning, we
treat computer code differently from the rest of your article in our production
process. To that end, we request separate files for computer code.
In Online Supplemental Material We request that runnable source code be included as supplemental material to
the article. For more information, visit Supplementing Your Article With Online
Material.
In the Text of the Article
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If you would like to include code in the text of your published manuscript, please
submit a separate file with your code exactly as you want it to appear, using
Courier New font with a type size of 8 points. We will make an image of each
segment of code in your article that exceeds 40 characters in length. (Shorter
snippets of code that appear in text will be typeset in Courier New and run in
with the rest of the text.) If an appendix contains a mix of code and explanatory
text, please submit a file that contains the entire appendix, with the code keyed
in 8-point Courier New.
Tables
Use Word's Insert Table function when you create tables. Using spaces or tabs
in your table will create problems when the table is typeset and may result in
errors.
Submitting Supplemental Materials
APA can place supplemental materials online, available via the published article
in the PsycARTICLES® database. Please see Supplementing Your Article With
Online Material for more details.
Abstract and Keywords
All manuscripts must include an abstract containing a maximum of 250 words
typed on a separate page. After the abstract, please supply up to five keywords
or brief phrases.
References
List references in alphabetical order. Each listed reference should be cited in
text, and each text citation should be listed in the References section.
Examples of basic reference formats:
Journal Article: Hughes, G., Desantis, A., & Waszak, F. (2013). Mechanisms of intentional
binding and sensory attenuation: The role of temporal prediction,
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temporal control, identity prediction, and motor prediction. Psychological
Bulletin, 139, 133–151. http://dx.doi.org/10.1037/a0028566
Authored Book: Rogers, T. T., & McClelland, J. L. (2004). Semantic cognition: A parallel
distributed processing approach. Cambridge, MA: MIT Press.
Chapter in an Edited Book: Gill, M. J., & Sypher, B. D. (2009). Workplace incivility and organizational
trust. In P. Lutgen-Sandvik & B. D. Sypher (Eds.), Destructive
organizational communication: Processes, consequences, and
constructive ways of organizing (pp. 53–73). New York, NY: Taylor &
Francis.
Figures
Graphics files are welcome if supplied as Tiff or EPS files. Multipanel figures
(i.e., figures with parts labeled a, b, c, d, etc.) should be assembled into one file.
The minimum line weight for line art is 0.5 point for optimal printing.
For more information about acceptable resolutions, fonts, sizing, and other
figure issues, please see the general guidelines.
When possible, please place symbol legends below the figure instead of to the
side.
APA offers authors the option to publish their figures online in color without the
costs associated with print publication of color figures.
The same caption will appear on both the online (color) and print (black and
white) versions. To ensure that the figure can be understood in both formats,
authors should add alternative wording (e.g., "the red (dark gray) bars
represent") as needed.
For authors who prefer their figures to be published in color both in print and
online, original color figures can be printed in color at the editor's and publisher's
discretion provided the author agrees to pay:
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$900 for one figure
An additional $600 for the second figure
An additional $450 for each subsequent figure
Permissions
Authors of accepted papers must obtain and provide to the editor on final
acceptance all necessary permissions to reproduce in print and electronic form
any copyrighted work, including test materials (or portions thereof), photographs,
and other graphic images (including those used as stimuli in experiments).
On advice of counsel, APA may decline to publish any image whose copyright
status is unknown.
Download Permissions Alert Form (PDF, 13KB)
Publication Policies
APA policy prohibits an author from submitting the same manuscript for
concurrent consideration by two or more publications.
See also APA Journals ® Internet Posting Guidelines .
APA requires authors to reveal any possible conflict of interest in the conduct
and reporting of research (e.g., financial interests in a test or procedure, funding
by pharmaceutical companies for drug research).
Download Disclosure of Interests Form (PDF, 38KB)
Authors of accepted manuscripts are required to transfer the copyright to APA.
For manuscripts not funded by the Wellcome Trust or the Research
Councils UK
Publication Rights (Copyright Transfer) Form (PDF, 83KB)
For manuscripts funded by the Wellcome Trust or the Research Councils
UK
Wellcome Trust or Research Councils UK Publication Rights Form (PDF,
34KB)
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Ethical Principles
It is a violation of APA Ethical Principles to publish "as original data, data that
have been previously published" (Standard 8.13).
In addition, APA Ethical Principles specify that "after research results are
published, psychologists do not withhold the data on which their conclusions are
based from other competent professionals who seek to verify the substantive
claims through reanalysis and who intend to use such data only for that purpose,
provided that the confidentiality of the participants can be protected and unless
legal rights concerning proprietary data preclude their release" (Standard 8.14).
APA expects authors to adhere to these standards. Specifically, APA expects
authors to have their data available throughout the editorial review process and
for at least 5 years after the date of publication.
Authors are required to state in writing that they have complied with APA ethical
standards in the treatment of their sample, human or animal, or to describe the
details of treatment.
Download Certification of Compliance With APA Ethical Principles Form
(PDF, 26KB)
The APA Ethics Office provides the full Ethical Principles of Psychologists and
Code of Conduct electronically on its website in HTML, PDF, and Word format.
You may also request a copy by emailing or calling the APA Ethics Office (202-
336-5930). You may also read "Ethical Principles," December 1992, American
Psychologist, Vol. 47, pp. 1597–161.
170
Appendix C: Instructions to authors: Main research project
Instructions for Authors
Journal of Cancer Survivorship
Types of Articles: Original Papers, Reviews, and Editorials.
Editorial procedure
Single-blind peer review
This journal follows a single-blind reviewing procedure. Authors are therefore
requested to submit a title page, containing title, all author names, affiliations,
and the contact information of the corresponding author. Any
acknowledgements, disclosures, or funding information should also be included
on this page.
Manuscript Submission
Submission of a manuscript implies: that the work described has not been
published before; that it is not under consideration for publication anywhere else;
that its publication has been approved by all co-authors, if any, as well as by the
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been carried out. The publisher will not be held legally responsible should there
be any claims for compensation.
Permissions
Authors wishing to include figures, tables, or text passages that have already
been published elsewhere are required to obtain permission from the copyright
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permission has been granted when submitting their papers. Any material
received without such evidence will be assumed to originate from the authors.
171
Online Submission
Authors should submit their manuscripts online. Electronic submission
substantially reduces the editorial processing and reviewing times and shortens
overall publication times. Please follow the hyperlink “Submit online” on the right
and upload all of your manuscript files following the instructions given on the
screen.
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The title page should include:
- The name(s) of the author(s)
- A concise and informative title
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Abstract
Please provide a structured abstract of 150 to 250 words which should be
divided into the following sections:
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Keywords
Please provide 4 to 6 keywords which can be used for indexing purposes.
172
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Smith JJ. The world of science. Am J Sci. 1999;36:234–5.
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Slifka MK, Whitton JL. Clinical implications of dysregulated cytokine production.
J Mol Med. 2000; doi:10.1007/s001090000086
Book
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Blenkinsopp A, Paxton P. Symptoms in the pharmacy: a guide to the
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Bourne GH, Danielli JF, Jeon KW, editors. International review of cytology.
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176
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177
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182
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Journal of Cancer Survivorship
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4301 Jones Bridge Road
Bethesda, Maryland 20814-4799
e-mail: [email protected]
183
Appendix D: Interview schedule: Service improvement project
1. Thank you for agreeing to take part in our research
2. Do you have any questions about what you’ve already read? Ready to go ahead?
3. These questions relate to your experiences of having a child with a life-limiting condition
4. The interview may take up to 40 minutes.
5. You don’t have to answer all of the questions.
6. You can ask questions as we go through.
Information about the child
How long ago was your child diagnosed with their condition?
How many professionals have you had contact with regarding your child’s
condition?
0-15 10-15 15-20 20+
Has your child had stays in hospital?
o How many admissions have they had?
o When was the last time they were in hospital? How long was their
stay?
Were any emergency admissions?
184
o How many?
o When was the last time that this happened?
Emotional and psychological support
What emotional or psychological support has been offered to you?
Did you take up the offer of this support? If so,
o Who provided this support? (Person and whether a professional or
friends/family)
o In what form did you receive this? E.g. telephone, face to face
o What was most helpful about this support?
o Was this sufficient, or would you have liked more?
If not, what stopped you taking up the offer of support?
o Did you ask for support from anyone?
o If not, why not, what would stop you asking for help?
What, if anything, would you have liked more of?
What other forms of support for emotional and psychological effects
would have been helpful?
o When would this have been most helpful to receive?
185
Information about emotional and psychological responses
Have you received any information about the potential impact of your
child’s illness on you?
o How did you get this information?
o What was helpful about it?
o Was it given at the right time point?/If not, when would it have
been useful?
o Is there any other information that would have been helpful to have
received?
o At what time point?
o Do you think some written information about the potential
emotional impact on you of your child’s illness would have been
helpful?
o What would be helpful to include?
End of interview
Debrief
186
Appendix E: Questionnaires: Main research project
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Appendix F: Description of questionnaire measures: Main research project
Mental defeat
The Pain Self Perception Scale (PSPS; Tang, Salkovskis, & Hanna, 2007) (24
items). The PSPS assesses perceptions of defeat associated with a recent
episode of intense pain. It features 24 items, which were adapted from the
Defeat Scale (Gilbert & Allan, 1998) and MDTS (Mental Defeat during Trauma
Scale; Dunmore, Clark, & Ehlers, 1999) that are rated for their applicability
during the pain episode. Items are rated on a 5-point scale (0 =‘‘Not at
all/Never,’’ 1 =‘‘Very little,’’ 2 = ‘‘Moderately,’’ 3 =‘‘Strongly,’’ 4 =‘‘Very strongly’’),
generating a total score with a possible range of 0 – 96. The PSPS has good
psychometric properties in pain patients (Tang, Salkovskis, & Hanna, 2007). The
measure was adapted for the present research to measure mental defeat in
cancer survivors in response to their experience of cancer. The following
statement was added; ‘Because of my experience of cancer and treatment…’.
Existential concerns
The Life Scheme subscale of the Spirituality Index of Wellbeing (SIWB;
Daaleman & Frey, 2004). The 6 item Life Scheme subscale of the SIWB was
used to measure existential concerns. Items are rates on a 5 point scale ranging
from 1 “strongly disagree” to 5 “strongly agree”. All items were reversed scored
to facilitate analysis compared to other measures, with higher scores indicating
greater existential concerns. The range of total possible scores is 6 – 30. The
subscale has good psychometric properties and is designed for use in quality of
life research in physical health populations (Daaleman & Frey, 2004).
207
Beliefs about emotions
Beliefs about Emotions Scale (BES; Rimes & Chalder, 2010) (12-items). The
BES is a 12-item scale that measures beliefs about the experience and
expression of emotions. Items are rated on a scale from 0 to 6, with higher total
scores indicating more maladaptive beliefs. The BES has demonstrated good
reliability, validity and sensitivity to change across a range of psychological
problems (Rimes & Chalder, 2010).
Intolerance of uncertainty
Intolerance of Uncertainty Scale-12 (IUS-12; Carleton, Norton & Adsmundson,
2007) (12 items). The IUS-12 is a recently developed 12 item version of the
original IUS, a 27-item measure of intolerance of uncertainty (Buhr & Dugas,
2002). It measures unacceptability of uncertainty and manifestations of
uncertainty similar to common anxiety symptoms. The IUS was validated for use
in English populations by Buhr and Dugas (2002), and found to have good
reliability and validity. It was shortened to a 12 item version by Carleton et al
(2007) with a two factor structure (prospective anxiety and inhibitory anxiety),
which improved psychometric properties. Items are rated on a scale from 1 ‘not
at all characteristic of me’ to 5 ‘entirely characteristic of me’, with a total scores
ranging from 27 – 135. Higher scores indicate higher levels of intolerance of
uncertainty. The scale has been shown to be valid and reliable in breast cancer
patients (e.g. Jones, Hadjistavropoulos & Gullickson, 2014).
Health anxiety
Health Anxiety Inventory Short Week (SHAI; Salkovskis, Rimes, Warwick &
Clark, 2002) (32 items). The SHAI is a reliable and valid measure of health
anxiety, and is the most sensitive to change over time of the HAI versions. The
first 14 items will be used to assess health anxiety symptoms. Items are rated on
208
a 4 point scale from 0 – 3, with a total score range of 0 – 42. Higher scores
indicate higher levels of health anxiety. A cut off score of 15 indicates elevated
health anxiety (Wright & Salkovskis, 2007), and a score of 18 or higher denotes
people who are likely to meet criteria for a diagnosable disorder (Rode,
Salkovskis, Dowd & Hanna, 2006; Seivewright et al, 2004). The anticipated
burden of illness is measured by 4 additional items. Avoidance and reassurance
seeking subscales will be used to assess reassurance seeking and avoidance
behaviour comprising 10 items and 8 items, respectively.
The SHAI has good psychometric properties in the general population
(Salkovskis et al., 2002; Abramowitz, Deacon & Valentiner, 2007) and has been
used in medical populations (Tyrer et al, 2014). The factor structure has been
found to be the same in medical and non-medical samples (Alberts, Sharpe,
Kehler & Hadjistavropoulos, 2011). The measure was adapted for the current
study with permission from the author to include the following statement; ‘Please
answer in relation to worries about your health in general (this may include
worries about cancer)’.
Quality of life
Quality of Life Index Cancer Version III (QLI-C III; Ferrans, 1990). The QLI-C is a
33-item measure of quality of life measuring satisfaction across a number of
domains. Part 1 (33 items) measures how satisfied the respondent is with areas
of their life on a scale of 1 ‘very dissatisfied’ to 6 ‘very satisfied’. Part 2 (33
items) measures how important each area is to the respondent on a scale of 1
‘very unimportant’ to 6 ‘very important’. The scale is centred on zero by
subtracting 3.5 from each satisfaction item. Satisfaction responses are weighted
with the paired importance responses. The sum of the weighted responses
produces an overall total score. To prevent bias due to missing data, each
respondent’s total score is divided by the number of items answered by that
individual. In order to eliminate negative numbers, 15 is added to each total
209
score to produce a total final score, with a range of 0-30 with higher scores
indicating better quality of life. The QLI-C has good reliability, validity and
sensitivity to change (Ferrans & Powers, 1985).
Psychological distress
Patient Health Questionnaire (PHQ-9; Kroenke, Spitzer & Williams, 2001) and
Generalised Anxiety Disorder Assessment (GAD-7; Swinson, 2006) (combined:
16 items). The combined PHQ-9 and GAD-7 were used to assess depression
and anxiety as a measure of psychological distress. The PHQ-9 total score
ranges from 0 to 27 with higher scores indicating higher levels of depression. A
total score for the seven items ranges from 0 to 21, with higher scores
representing higher levels of anxiety. The PHQ-9 and GAD-7 have been used to
screen for depression and anxiety in cancer patients, and have good
psychometric properties (Thekkumpurath et al, 2011; Spitzer et al, 2006).
210
211
212
213
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215
216
217
218
219
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Appendix H: Z test comparisons with normative data: Main research project
Comparisons with normative data
Mean scores for each measure at T1 (n=90) and T2 (n=69) are reported in the
following sections. A series of z-tests for means were conducted comparing the
means for the total sample at T1 (n=90) with normative data for each measure.
Effect sizes are reported as measured by Cohen’s d (Cohen, 1992).
Mental defeat
The mean mental defeat score at T1 was 14.5 (SD=15.8, range=0-66), and 12.3
(SD=15.4, range=0-68) at T2. The T1 mean score was significantly higher than
normative data for pain-free controls (M= 7.2, SD=9.3, n=79) (z = 7.41, p =
0.0001, two-tailed). The magnitude of this difference was large (d=0.78). There
was no significant difference between the T1 sample mean and normative data
for patients with acute pain (M = 14.6, SD= 17.4, n=38) (z = -0.76, p=0.94, two
tailed, d= -0.01). The mean was significantly lower than normative data for
chronic pain patients (M= 36.2, SD=28.9, n=94) (z= -7.14, p=0.0001, two tailed).
The magnitude of this difference was large (d= -0.75). There was no significant
difference between the T1 sample mean and normative data for community
volunteers with chronic pain (M=16.7, SD=19.3, n=32) (z= -1.10, p=0.27 two
tailed, d= -0.12).
Existential concerns
The mean existential concerns score for the total sample at T1 was 13.6
(SD=6.2, range= 6 – 30) and 14.2 (SD=6.3, range= 6 – 30) at T2. The T1
sample mean (M = 13.6, SD=6.2) was compared to normative data for
outpatients at primary care clinics (M=24.58, SD=4.97, n=523) for The Life 221
Scheme subscale of the Spirituality Index of Wellbeing (SIWB; Daaleman &
Frey, 2004). The mean for the current sample was significantly lower than
normative data (z= -20.96, p=0.0001, two tailed), with a large effect size (d= -
2.21), meaning the current sample had less existential concerns.
Beliefs about emotions
The mean beliefs about emotions score for the total sample at T1 was 35.0
(SD=15.6, range= 0 - 68). The mean at T2 was 31.7 (SD=14.9, range= 0 - 72).
The T1 sample mean (M = 35.0, SD=15.4) was compared to original normative
data for healthy controls and patients with CFS for the Beliefs About Emotions
Scale (BES; Rimes & Chalder, 2010). Compared to normative data for healthy
controls (M=27.9, SD=11.3, n=73), the current sample scored significantly
higher (z= 5.96, p=0.0001, two tailed), indicating higher levels of beliefs about
the unacceptability of experiencing or expressing negative emotions. This
yielded a medium effect size (d=0.62). The T1 sample mean was comparable to
normative data for patients with chronic fatigue syndrome (M = 35.0, SD=14.3,
n=121) (z= 0.0001, p=1.00, d=0.0001).
Intolerance of uncertainty
The mean intolerance of uncertainty score at T1 was 29.8 (SD=8.7, range=14 –
55), and 27.8 (SD=8.7, range= 16 – 60) at T2. The mean at T1 (M = 29.8,
SD=8.7) was compared to original normative data firstly for healthy University
students (Carleton, Norton & Asmundson, 2007) and recent normative data for
patients with Generalised Anxiety Disorder (Khawaja & Yu, 2010). The 27-item
IUS has been used with cancer patients, whereas the IUS-12 has not.
Compared to normative data for University students (M=25.85, SD=9.45,
n=818), the current sample scored significantly higher (z= 3.97, p=0.0001, two
tailed), approaching a medium effect size (d=0.42). Conversely, compared to
222
normative data for patients with GAD (M= 36.76, SD=8.72, n=50), the current
sample mean was significantly lower (z= -7.57, p=0.0001), with a large effect
size (d= -0.80).
Health anxiety
The mean health anxiety score (items 1 – 14) for the total sample at T1 was
15.3 (SD=6.7, range= 0 – 35), and 14.3 (SD=6.9, range= 0 – 31). A cut off score
of 15 indicates elevated health anxiety and a score of 18 or higher denotes
people who are likely to meet criteria for a diagnosable disorder. The mean is
above 15, and 52.2% of the sample scored 15 or over, with a further 38.9%
scoring 18 or over.
Means for the current sample were compared with the original normative data
for the first 14 items of the Short Health Anxiety Inventory (SHAI; Salkovskis,
Rimes, Warwick & Clark, 2002). The current sample scored significantly lower
than patients with a diagnosis of hypochondriasis in this study (M=30.1, SD=5.5,
n=24) (z= -25.53, p=0.0001, d= -2.69). Data for the current sample was
comparable to anxious controls (M=14.9, SD=6.2, n=19) (z=0.61, p=0.0001,
d=0.06), and significantly higher than healthy controls (M=9.4, SD=5.1, n=159)
(z= 10.97, p=0.0001, d=1.16).
Quality of life
The mean quality of life score for the total sample at T1 was 21.9 (SD=4.1,
range=13.5 – 29.0) and 22.5 (SD=4.1, range=13.4 – 29.1) at T2. Compared to
the initial validation study with breast cancer patients (Ferrans, 1990), the mean
quality of life score at T1 was significantly lower (M=23.03, SD=4.62, n=111) (z=
-2.32, p=0.020, d= -0.24), meaning the current sample reported significantly
poorer quality of life than reported in normative data.
223
Psychological distress
The mean distress score for the total sample at T1 was 12.6 (SD=9.6, range= 0
- 40), and 10.9 (SD=9.6, range=0 – 37) at T2. The mean T1 depression score
was 6.8 (SD=5.6, range=0 – 23), and 5.9 (SD=5.2, range= 0 – 21) at T2.
Compared to normative data for the general population (M = 2.9, SD= 3.5,
n=5018; Kocalevent, Hinz and Brahler, 2013), the current sample scored
significantly higher (z=10.57, p=0.0001, two tailed), with a large effect size
(d=1.11).
In terms of severity of depression, 30% of participants scored above caseness
(>10; Kroenke & Spitzer, 2002), 46.7% below the cut off for mild depression (0-
4), 23.3% within the mild range (5-9), 18.9% in the moderate range (10-14), 10%
within the moderate-severe (15-19) range and 1.1% within the severe range
(>20) (Kroenke, Spitzer & Williams, 2001).
The mean anxiety score for the total sample at T1 was 5.8 (SD=4.7, range= 0 –
19) and 5.0 (SD=4.9, range= 0 – 18). Compared to normative data for the
general population (M = 2.95, SD=3.4, n=5030; Löwe et al, 2008), the current
sample scored significantly higher (z=7.95, p=0.0001, two tailed). The
magnitude of this effect was large (d=0.83). Thirty percent of participants scored
above caseness (>8; Kroenke et al, 2002, 2007), 45.6% scored below the cut off
for mild anxiety (<5), 33.3% within the mild range (5 – 9), 15.6% within the
moderate range (10 – 14) and 5.6% within the severe range (15 – 21) (Spitzer,
Kroenke, Williams & Lowe, 2006).
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