Health and Social Care in the Community (2016) 24(1), 12–26

doi: 10.1111/hsc.12184

Review Families’ and healthcare professionals’ perceptions of healthcare services for children and young people with medically unexplained symptoms: a narrative review of the literature Denise Hinton

BA MA PhD

and Susan Kirk

BNurs MSc PhD

School of Nursing, Midwifery and Social Work, University of Manchester, Manchester, UK Accepted for publication 29 October 2014

Correspondence Susan Kirk School of Nursing, Midwifery and Social Work University of Manchester Oxford Road, Manchester M13 9PL, UK E-mail: [email protected]

What is known about this topic

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Medically unexplained symptoms (MUS) in children and young people are a common but under-researched health issue. MUS can have a detrimental impact on children’s and young people’s functioning and well-being and they may require appropriate support to manage their illness. Considerable healthcare resources may be expended on this group and it is important that services meet children’s and young people’s support needs.

What this paper adds

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This paper is the first attempt to systematically review and synthesise families’ and healthcare professionals’ perceptions of healthcare services for children and young people with MUS. It highlights families’ and professionals’ ambivalence about current services with families reporting dissatisfaction with the approach of some HCPs. It suggests that the lack of wellconducted studies on this topic may act as a barrier to the delivery of appropriate evidence-based healthcare that meets the needs of families and young people.

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Abstract Children and young people frequently report physical complaints that have no observable physical pathology known as medically unexplained symptoms (MUS). Research suggests that MUS are associated with substantial physical and psychological impairments and may have a negative impact on children’s and young people’s functional status and well-being in the long term. Due to the potentially complex needs of this group, children and young people with MUS may require timely access to suitable health and social care services to effectively manage symptoms and achieve their academic, social and personal potential. Families and professionals can offer important insights into the availability and appropriateness of current community and specialist health and social care services. This review is the first critical evaluation and synthesis of research that has examined families’ and healthcare professionals’ (HCP) perceptions of healthcare services for children and young people with MUS. A systematic search of electronic databases and manual searches of key journals and reference lists identified 17 papers from 15 studies for inclusion in the review. The review highlights the paucity of rigorously conducted research on this topic. Studies have been narrowly focused on the views of a homogeneous group of mothers and young people attending single centres. There has been some attempt to examine doctors’ views, but the perceptions of children, fathers and health and social care professionals are absent or under-represented, and multi-site and longitudinal studies are lacking. Thematic analysis of the results from the included studies suggests that knowledge, communication, health beliefs and healthcare settings are factors that influence families’ and HCPs’ perceptions of services. Families report dissatisfaction with some HCPs’ approach to managing MUS. The findings suggest that children and young people with MUS are at risk of receiving suboptimal care and support because there is insufficient research to inform high-quality, evidence-based practice. Keywords: children, functional syndromes, healthcare professionals, medically unexplained symptoms, somatic disorders, young people

© 2015 John Wiley & Sons Ltd

Healthcare services for young people with MUS

Background Healthcare professionals (HCPs) use the term ‘medically unexplained symptoms’ (MUS) to describe reported physical complaints that have no detectable organic pathology (Geist et al. 2008). An estimated 10%–25% of children and young people report chronic somatic complaints, but only a small proportion of these symptoms will have an identifiable medical cause (Zuckerman et al. 1987, Hyams et al. 1996, Egger et al. 1999, Domenech-Llaberia et al. 2004, Carson et al. 2011). Commonly reported symptoms include headache, backache, fatigue, faintness, muscle pain, nausea and abdominal pain (Perquin et al. 2000, Bhatia et al. 2008, Vila et al. 2009). Although many HCPs report caring for children and young people with MUS (Eminson 2007, Geist et al. 2008), estimates need to be treated with caution as there is no agreed definition of what constitutes MUS and interpretations of the term may differ between medical specialties. This lack of clarity makes it difficult to conduct robust epidemiological studies of MUS and accurately calculate prevalence in different patient populations. MUS in children and young people appear to be complex and poorly understood (Eminson 2007). Research suggests that MUS are associated with multiple factors including parental illness, poverty, family dysfunction and bullying (Perquin et al. 2000, Eminson 2007, Schulte & Petermann 2011, Hansen et al. 2012, Van Den Eede et al. 2012). Although the term MUS is ostensibly neutral and does not imply a cause, the absence of identifiable organic aetiology may lead HCPs to advance a psychological interpretation of symptoms. MUS are frequently associated with dissociative/conversion disorders and functional somatoform disorders (Eminson 2007, Geist et al. 2008), although there is always the risk that there may be an organic explanation that has not been detected (cf. Spratt & Thomas 2008). MUS are also the main feature of named syndromes such as chronic fatigue syndrome (CFS) and myalgic encephalomyelitis (ME) that have no detectable pathological basis. Children and young people with MUS may have extensive and complex healthcare needs. Research suggests this group of young people are at increased risk of co-morbid psychopathology (Taylor & Garralda 2003, Konijnenberg et al. 2006, Crawley et al. 2009, Knook et al. 2011, Bould et al. 2013) and functional impairment in everyday activities with symptoms affecting sleep, school attendance and social activities (Smith et al. 2003, Konijnenberg et al. 2005, Sankey et al. 2006, Crawley & Sterne 2009). © 2015 John Wiley & Sons Ltd

Children’s symptoms may have a detrimental impact on family income and psychological health (Missen et al. 2012), and there is evidence to suggest that MUS in childhood is linked to poor health in adulthood, highlighting the long-term healthcare needs of this group (Campo et al. 2001, Walker et al. 2010). Research indicates that the investigation of MUS in children and young people consumes considerable healthcare resources owing to the frequent utilisation of services, specialist consultations and numerous investigations and treatments (Rosendal et al. 2004, Ibeziako & Bujoreanu 2011). Children and young people require access to appropriate and timely support to manage symptoms and minimise distress and functional impairment. However, there is some evidence to suggest that clinicians find it challenging to care for children and young people with MUS due to the lack of guidelines and diagnostic criteria for unlabelled symptoms apart from CFS (NICE 2007) (Dell & Campo 2011) and a perceived reluctance among young people and parents to consider psychosocial explanations for physical symptoms (Christie & Viner 2005, Geist et al. 2008). Furthermore, research in adult healthcare settings suggests that HCPs are uncertain how to support patients with MUS and feel under pressure to order further investigations to ‘rule out’ organic disease (Hartz et al. 2000, Wileman et al. 2002, Ring et al. 2004). Although clinical trials of intervention programmes for children and young people with MUS are lacking, experienced clinicians recommend HCPs seek to engage families in a therapeutic relationship to successfully manage the adverse effects of unexplained physical symptoms in children and young people (Campo & Fritz 2001, Garralda & Chalder 2005). This requires HCPs to work in partnership with parents and young people (Gabe et al. 2004) to gather accurate accounts of the child’s symptoms, particularly if the child is unable to recall or articulate their symptoms (Taylor et al. 1996), identify contextual factors that have influenced the onset of symptoms (Dell & Campo 2011) and encourage families to participate in psychological therapies (Kozlowska et al. 2013). Furthermore, a multidisciplinary approach is advocated to manage the complex needs of young people (Campo & Fritz 2001), although Robins et al. (2005) contend that referrals to child and adolescent mental health services are often a last resort when other approaches have failed. In addition, clinicians are advised to exercise caution during the investigation of MUS. Performing a battery of tests to search for and exclude physical

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pathology is discouraged to avoid iatrogenic harm and the over-medicalisation of symptoms, which, in turn, may inadvertently encourage families to seek an organic explanation (Campo & Fritz 2001, Geist et al. 2008). However, this may be difficult to achieve in practice. Kreipe (2006) argues that a reliance on diagnostic tests, particularly magnetic resonance imaging and other technology advanced tools, to explore reported symptoms promotes a dualistic conceptualisation of the body. Kreipe (2006) contends that this duality may be unhelpful to the young person, their family and the HCP because this advances the notion that the body and mind are distinct and unconnected entities when physical and psychological health may be closely linked. Consequently, families may feel frustrated by the lack of progress in detecting a physical cause while resisting psychological explanations for symptoms. The current evidence suggests that children and young people with MUS may require access to suitable and timely support from specialist and community-based services to effectively manage symptoms and any associated physical and/or psychological difficulties. Due to the frequency and complexity of MUS in children and adolescents, the adverse implications of MUS on young peoples’ functional status, well-being and long-term health, and the considerable healthcare resources expended, it is important to ascertain if current services are meeting children’s and young people’s support needs. Families and professionals can offer important insights into the availability and appropriateness of current services. The purpose of this review is two-fold. First, it aims to provide a comprehensive synthesis of existing research that examines families’ and HCPs’ perceptions of healthcare services for children and young people with MUS. Second, it aims to evaluate the quality and breadth of available literature on this topic, highlight gaps in the current knowledge base, and identify areas for further research, with a view to informing the planning of future healthcare services for children and young people with MUS.

Methods A comprehensive narrative review of the literature was conducted to identify, critically evaluate and synthesise studies that examined families’ and HCPs’ perceptions of healthcare services for children and young people with MUS. A systematic search of computerised databases with no date restrictions was carried out using a range of search terms including named syndromes with MUS (e.g. ME) and conditions which are psychological in nature (e.g. conver14

sion disorder). Table 1 presents the search strategy and inclusion/exclusion criteria. This was necessary due to the overlap between definitions and biopsychosocial dimensions to reported symptoms, reflecting the complexity of conceptualising MUS (Eminson 2007). In total, 1161 records were identified for potential inclusion by the electronic database search (see Figure 1). Most of these records were excluded from the review because they focused exclusively on the presentation, clinical management and aetiology of MUS in children and young people. Each title was reviewed by the first author to assess eligibility and the abstracts of 244 papers were screened to identify if they met the study inclusion criteria. Following assessment of the abstracts, full copies of 28 papers were retrieved and 17 papers from 15 empirical studies were included in the review. The study selection process is shown in Figure 1. Five additional papers were identified during the additional searching process described above. The quality of all included studies was assessed using criteria available from the United Kingdom Centre for Reviews and Dissemination (CRD 2008) and the Critical Appraisals Skills Programme (CASP 2010). The methodological strengths and limitations of each study are highlighted in Table 2, but no papers were excluded on the basis of quality assessment. A summary of each paper is included in Table 2. Thematic analysis of the papers was used to identify patterns and themes to describe the data (Braun & Clarke 2006). All papers were reviewed and coded by the first author, and study findings were coded and then categorised into themes to explore connections within and between papers. As analysis continued, new categories emerged and existing categories were modified to generate a detailed and coherent synthesis of the data.

Results In total, 17 papers from 15 empirical studies were included in the review. The papers describe families’ and professionals’ perceptions of primary and secondary healthcare services in the United Kingdom (UK) (n = 11), the Netherlands (n = 3), Ireland (n = 1), Norway (n = 1) and the United States (US) (n = 1). No papers relevant to the review were identified from low- and middle-income countries. Papers focused on the following symptoms or labelled conditions: unexplained pain (n = 7), CFS/ME (n = 7) and MUS (n = 3). The review includes seven qualitative studies, six quantitative studies and two mixedmethods studies. © 2015 John Wiley & Sons Ltd

Healthcare services for young people with MUS

Table 1 Literature search strategy Medically unexplained symptoms literature search Databases searched ASSIA British Nursing Index CINAHL plus Medline Psych Info SAGE full-text collections Hand searching Clinical Child Psychology and Psychiatry Child and Adolescent Mental Health Grey literature Unpublished papers Limitations No date restrictions; human subjects; English language Inclusion criteria Empirical research studies and literature reviews that focus on Children and young people aged 0–18 years old reporting physical complaints with no identifiable organic cause including the following named syndromes: chronic fatigue syndrome (CFS), myalgic encephalopathy (ME), post-viral fatigue syndrome and irritable bowel syndrome (IBS) The parents/carers of children with medically unexplained symptoms (MUS) HCPs’ knowledge and experience of supporting children and young people with MUS in primary or secondary care healthcare settings Exclusion criteria Empirical studies that focused exclusively on The presentation, clinical management, aetiology and/or prevalence of MUS in children and young people Children and young people’s experiences of symptoms (e.g. chronic pain) attributable to diagnosed conditions such as rheumatoid arthritis, for example. Clinical trials of medications and/or interventions to alleviate reported MUS in children and young people. Parents’ medically unexplained symptoms Adults with named syndromes such as CFS, ME, post-viral fatigue syndrome, fibromyalgia or IBS

Search strategy (combination of free text terms in title or abstract) Free text terms Medically unexplained symptom* OR medically unexplained physical symptom* OR unexplained symptom* OR somatic disorder* OR somatic pain disorder OR somatisation OR somatoform disorder* OR somatisation disorder* OR polysymptomatic somatisation OR conversion disorder* OR polysymptomatic conversion disorder* OR functional syndrome* OR functional somatic syndrome* OR psychosomatic complaint* OR psychosomatic disorder* OR dissociative disorder* OR pervasive refusal syndrome* OR chronic pain OR chronic pain disorder OR somatic pain OR somatic pain disorder OR chronic fatigue syndrome OR myalgic encephalopathy OR post viral fatigue syndrome AND Child* OR adoles* OR paediatric OR pediatric OR young people OR young person OR youth OR teenager

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HCP, healthcare professional.

The quality of the included papers varied considerably and there were notable limitations to the available data. The majority of data were collected from single sites and focused on the views of young people (aged 12 years old and over), mothers and doctors. The views of children (aged 11 years old and younger) were absent from the data (although one study recorded pain and functional impairment by proxy) and the perceptions of fathers and other professionals were under-represented. Quantitative data were collected via cross-sectional surveys using instruments that were not validated or retrospective reviews of medical records. No multi-site or longitudinal studies have been conducted. © 2015 John Wiley & Sons Ltd

Qualitative data were based on homogeneous participant samples (typically white and female) and several studies recruited very small numbers of participants. The description of qualitative data analysis procedures varied between studies and the potential for research bias was not always addressed satisfactorily. Five of the included studies were conducted more than 10 years ago and it is not clear whether the findings of these studies are relevant to current healthcare services. Despite variation in the quality of the included studies, four key themes were identified during synthesis of the study findings: knowledge, communication, health beliefs and healthcare settings. These are discussed below. 15

D. Hinton & S. Kirk

Titles idenƟfied and screened n = 1161 Excluded n = 916 Abstracts screened n = 245 Excluded n = 217 Full copies retrieved and assessed for eligibility n = 28

Excluded n = 16 Focused on generic chronic pain in children Described methods to elicit children’s opinions about pain Described paƟents’ quality of life or health idenƟty DescripƟon of the disorder and diagnosƟc assessment Described impact on family members Case report

n=1 n=3 n= 3 n=2 n=1 n=6

Papers that met the inclusion criteria n = 12

Included n = 5 Hand search n=0 Reference lists n=5

Papers included in the review n = 17

Figure 1 Flow chart of paper selection process.

Knowledge Two small UK qualitative studies suggest that young people and parents feel that HCPs are failing to provide families with sufficient levels of information, understanding and support during medical encounters for children and young people experiencing unexplained symptoms (Dennison et al. 2010, Jelbert et al. 2010). Families reported difficulty accessing experienced and knowledgeable HCPs in primary and secondary healthcare services resulting in delays to diagnosis and referral to specialist services (Jelbert et al. 2010, Webb et al. 2011, Beasant et al. 2014). Young people report receiving limited advice about how to effectively manage symptoms (Jelbert et al. 2010). Interviews with 13 mothers with a child with a diagnosis of CFS/ME in the UK suggested parents needed to be proactive and persistent to gather information about the condition, and identify and negotiate access to a specialist CFS/ME service (Beasant et al. 2014). Families viewed specialist services for conditions such as CFS positively because it enabled 16

access to knowledgeable HCPs (Dennison et al. 2010, Beasant et al. 2014). Similarly, a UK mixed-methods study suggests HCPs are concerned that they lack sufficient knowledge and expertise to care for this group. In a survey of 128 paediatric HCPs working in one UK hospital, respondents reported feeling that children and young people with MUS have unmet support needs (Glazebrook et al. 2009). In subsequent qualitative interviews with 12 survey respondents, HCPs reported feeling ambivalent about current service provision. HCPs reported feeling that children and young people have complex and demanding needs, but suggested they were uncertain how best to deliver appropriate care to families on busy hospital wards (Furness et al. 2009). In particular, HCPs appeared to feel that a lack of time and training prevented them from delivering tailored and specialised care (Furness et al. 2009). Furthermore, the absence of established procedures for managing MUS in children and young people appears to increase the potential for professional © 2015 John Wiley & Sons Ltd

Focus of the study

Interventions for CFS/ME UK

Experiences of chronic pain in childhood UK

Parents’ perception of severe recurrent abdominal pain UK

Interventions for CFS/ME UK

Providing care for children and young people with MUS UK

Author and year

Beasant et al. (2014)

Carter (2002)

Crushell et al. (2003)

Dennison et al. (2010)

Furness et al. (2009)

Table 2 Papers included in the review

In-depth interviews with 13 mothers and 12 adolescents (aged 12– 18 years old) participating in a feasibility study of interventions for CFS/ME Interview topic guide presented, study limitations highlighted, implications for practice and further research discussed Purposive sampling from one centre only, ethical issues not discussed, limited discussion of analysis procedure, unclear how thematic codes were assigned to data Three young people (aged 12–13 years old) with chronic pain, and their parents, completed a journal over a period of 6 weeks and took part in a minimum of two interviews Mothers and fathers participated, families able to comment on interview transcripts, novel methods Small purposive sample from one centre only, sample not diverse, unclear how themes were derived from journals and interviews, potential for research bias not discussed The study was conducted in two parts: (i) a review of the medical records of 28 children with recurrent abdominal pain; (ii) a structured telephone interview with 28 parents Brief description of data analysis procedure, inferential statistics reported Small sample size, questionnaire not presented, few fathers, age range of children not reported Semi-structured interviews with 16 young people (aged 16– 24 years old) and 16 parents participating in a trial of CBT versus psycho-education Response rate given, inductive thematic analysis of data and coding strategy described, copy of interview guide presented Purposive sampling from one centre only, lack of diversity in sample (majority white female adolescents), few fathers Part of a mixed-methods study. This paper reported the findings of interviews and focus groups with 12 paediatric health professionals (following a survey of HCPs; see Glazebrook et al. 2009 below) Participants recruited from diverse medical specialities. Study limitations and areas for further research highlighted Purposive sampling from one site only, data analysis procedure and potential researcher bias not described

Methodology and sample (including quality appraisal)

© 2015 John Wiley & Sons Ltd HCPs reported feeling they lacked sufficient time and expertise to meet the needs of children and young people with MUS. HCPs requested further training and support

Parents and young people reported feeling HCPs lacked sufficient understanding of their condition prior to attending specialist therapy. Parents and young people were positive about attending therapy, but expressed ambivalence about the setting of the intervention

Parents of children with ongoing pain were more likely than parents whose children had recovered to believe the cause of the pain was organic only. The majority of parents of children with ongoing pain rated the input of a gastroenterologist helpful, but did not find the involvement of a psychiatrist useful

Parents reported difficulty obtaining a diagnosis for their child and young people reported feeling their own opinions were ignored or dismissed by HCPs

Participants reported difficulty accessing specialist services for CFS/ME. Parents and young people appeared to view specialist care positively

Main findings

Healthcare services for young people with MUS

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Focus of the study

Providing care for children and young people with MUS UK

Parents’ perception of adolescents chronic pain USA

Experiences of chronic fatigue syndrome Norway

Mothers’ perception of childhood chronic pain The Netherlands

Experiences of chronic fatigue syndrome UK

Author and year

Glazebrook et al. (2009)

Guite et al. (2009)

Hareide et al. (2011)

Hunfeld et al. (2002)

Jelbert et al. (2010)

Table 2 (continued)

Participants reported on the demanding nature of care and perceived children as having unmet support needs. Participants would like to receive further support from CAMHS

Self-reported survey to assess HCPs’ attitudes to caring for children with MUS. Completed by 128 HCPs; 50.4% response rate Data analysis procedure described and statistical significance reported Questionnaire piloted, but not validated prior to use. Not clear if the sample is representative of the target population. Few allied health professionals participated Retrospective review of records and psychological questionnaire data from 138 patients between the ages of 13 and 18 years who received the paediatric multidisciplinary pain clinic’s standard initial assessment questionnaire battery during a 2 ½ year period Data analysis procedure described and inferential analyses reported Survey not validated (adapted from other measures), fathers under-represented Semi-structured, in-depth interviews conducted with nine adolescent patients (12–17 years old) awaiting diagnosis for CFS/ ME Interview guide was informed by existing literature in this area and empirical research on the adult population Small purposive sample from one centre, potential research bias and interaction between researcher and researched not discussed Survey of mothers of 77 children (aged 5–11 years old) to assess the child’s functional status and impact of chronic pain on the family. Demographic data also collected. Response rate 17.5% Data analysis procedure described and inferential statistics recorded. Sample representative of study population Proxy assessment of child’s pain and functional disability, no copy of questionnaire provided, no fathers Semi-structured interviews with five adolescents (aged 13– 18 years old) who were considered to have recovered from CFS Interview topic guide developed in conjunction with professionals and voluntary organisation, data analysis procedure described and cross-validation of data conducted Small purposive sample from one centre, sample not diverse (all white British, four females) Participants attributed CFS to a physical cause. Participants reported diagnostic delay and a perceived lack of understanding of the condition among HCPs

Reported pain was mild and did not adversely affect the child’s functional status or school attendance. Children’s pain had a negative impact on family life

All participants attributed the disease to somatic causes. Participants reported experiencing considerable delays in receiving a diagnosis and negative attitudes from HCPs

Parents’ beliefs and worries about adolescent experience of chronic musculoskeletal pain are associated with the adolescent’s pain-related disability, family functioning and parenting stress

Main findings

Methodology and sample (including quality appraisal)

D. Hinton & S. Kirk

© 2015 John Wiley & Sons Ltd

© 2015 John Wiley & Sons Ltd

Providing care for children and young people with unexplained chronic pain The Netherlands

Providing care for children and young people with unexplained chronic pain The Netherlands

Providing care for children and young people with functional abdominal pain UK Experiences of MUS UK

Providing care for children and young people with CFS UK

Konijnenberg et al. (2004)

Konijnenberg et al. (2006)

Lindley et al. (2005)

Richards and Smith (1998)

Morris and Ogden (2012)

Focus of the study

Author and year

Table 2 (continued)

Diagnostic consensus could not be reached for over one-third of children (37%, n = 50) and paediatricians could not reach consensus on the primary cause of the symptoms in more than half of the patients (54%, n = 72)

Seventeen paediatricians assigned to three panels reviewed the full records for 134 patients aged 8–18 years old referred for pain of ≥3-month duration without a satisfactory explanation at presentation. Main outcome measures were agreement regarding the presumed primary cause and diagnostic approach for children with UCP Data analysis procedure described and inferential analyses reported Small sample of paediatricians with an interest in psychosomatic medicine Survey of 16 paediatricians to identify the intake characteristics of patients with unexplained chronic pain that were deemed diagnostically important for psychiatric morbidity Three researchers independently screened the paediatricians’ answers to identify major domains of information. Data analysis procedure described in detail and inferential statistics are reported Small purposive sample, unclear if representative of population Retrospective analysis of a cohort of 23 children aged 12 months after onset Small sample from one centre, data analysis procedure unclear, limitations not discussed Semi-structured interviews with 13 mothers of children aged 4– 16 years old who experienced somatic symptoms for which they had sought medical help at least once and received no clear diagnosis Some demographic information included Small snowball sample, limited description of methods and analysis procedure poorly described, limitations and implications for practice not discussed, no fathers Self-completed survey to assess 225 general practitioners’ experience, views concerning aetiology, and treatment preferences in patients under 19 years old with CFS; response rate 68% Demographic data collected Participants working in one specific health authority in England, instrument not validated, data analysis procedure not described, raw percentages only, no copy of the questionnaire published, men under-represented in sample The majority of respondents regarded CFS as a combined physical and psychological disorder and agreed that further medical opinion should be sought

Poor outcome was associated with a refusal to engage with psychological services, involvement of more than three consultants, lodging of a manipulative complaint with hospital management by the child’s family and lack of development of insight into psychosocial influences on symptoms Participants reported feeling that their child’s symptoms had an organic cause and described the negative effect of the symptoms on family life

Paediatricians appear to be aware of the signals to detect early psychiatric morbidity in children with unexplained chronic pain; age, social problem indicators, family structure, parental somatisation and school problems

Main findings

Methodology and sample (including quality appraisal)

Healthcare services for young people with MUS

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20

Experiences of CFS/ME Webb et al. (2011)

HCP, healthcare professionals; CFS/ME, chronic fatigue syndrome/myalgic encephalomyelitis; MUS, medically unexplained symptoms; CAMHS, child and adolescent mental health services; UCP, unexplained chronic pain.

The majority of participants (19 parents and 16 young people) reported organic causes. Some young people and their parents considered psychological symptoms important and psychiatric and psychological treatments helpful. Young people wanted professionals to listen to their experiences

Semi-structured interview with 21 young people (aged 11–20 years old) with a diagnosis of CFS/ME and their parents Data analysed thematically by the authors and discussed with a multidisciplinary team to minimise bias, justifies inclusion of selected quotes, acknowledges limitations Sample from one urban area, key sample characteristics missing including ethnicity of participants and gender of parents Mixed methods including assessment of 405 children with CFS/ME and semi-structured interviews with nine parents of children with CFS/ME to explore barriers to accessing healthcare Topic guide developed in conjunction with voluntary organisation, data analysis described in sufficient detail, themes discussed with HCPs and voluntary organisations Small sample from one specialist centre, few fathers Experiences of CFS Richards et al. (2006)

Few children with CFS/ME were seen within NICErecommended timeframes. Parents identified barriers to accessing specialist healthcare services including their own and healthcare professionals lack of knowledge about CFS/ME, negative attitudes towards the condition and difficulties communicating an invisible illness that they were not experiencing

Main findings Methodology and sample (including quality appraisal) Focus of the study Author and year

Table 2 (continued)

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disagreement and this may contribute to inconsistencies in the support offered to children and young people with MUS. Richards and Smith’s (1998) survey of 225 UK GPs suggests professional opinion may be divided over the aetiology and appropriate management of CFS. Similarly, a quantitative study of 17 paediatricians in The Netherlands by Konijnenberg et al. (2004) highlights the difficulties of reaching diagnostic and therapeutic consensus for children with unexplained chronic pain. Although there is evidence to suggest HCPs are aware of the key signals to identify risk of early psychiatric morbidity in children and young people with MUS, Konijnenberg et al. (2006) contend that in practice, this issue is managed without formal guidance. There is some limited qualitative evidence to suggest that HCPs would like additional input and support from child and adolescent mental health services (CAMHS) (Furness et al. 2009). There is variation in the quality of the studies included in this body of work, however, with qualitative studies narrowly focused on families’ perceptions of services for young people with CFS/ME and only one study examining clinicians’ perspectives of supporting children with MUS. Few of the qualitative studies fully describe the analysis procedure or findings and it is unclear how reported themes are derived from the data. It is also not clear if quantitative data are derived from representative samples or if findings can be extrapolated to HCPs’ working in other healthcare settings, with one study recruiting just 16 participants with a specialist interest in psychosomatic medicine (Konijnenberg et al. 2006), and another not describing the analysis procedure or reporting inferential statistics (Richards & Smith 1998). Communication Poor communication between HCPs and families appears to be a considerable problem. Young people and their parents reported feeling that their concerns were judged and dismissed by HCPs during medical encounters (Carter 2002, Dennison et al. 2010, Jelbert et al. 2010, Hareide et al. 2011, Webb et al. 2011, Morris & Ogden 2012). A qualitative study of 21 UK young people with a diagnosis of CFS/ME suggested that they wanted HCPs to listen to their experiences, although this did not always happen in practice (Richards et al. 2006). In one qualitative study of nine young people with CFS/ME in Norway, young people reported feeling that their symptoms were not taken seriously by HCPs and that HCPs attributed their complaints to psychological factors (Hareide © 2015 John Wiley & Sons Ltd

Healthcare services for young people with MUS

et al. 2011). This may have adverse implications for subsequent interactions in healthcare settings with young people reporting a reluctance to explain their pain to HCPs (Carter 2002) and experiencing anxiety about commencing specialist therapy (Dennison et al. 2010). The included studies suggest it may be challenging to orchestrate successful three-way (triadic) interactions between children, parents and professionals to discuss symptoms openly and without judgement (Carter 2002, Webb et al. 2011). Depending on children’s and young people’s ability and willingness to recall and describe their symptoms, and HCPs’ willingness to let children and young people contribute meaningfully to consultations, HCPs may focus on parents’ second-hand accounts of the presenting complaint(s) during consultations. However, semi-structured interviews with nine UK parents with a child with CFS/ME suggested that parents encountered difficulties communicating an invisible illness they were not experiencing themselves (Webb et al. 2011). A qualitative study of three young people aged 12–13 years with unexplained chronic pain, and their parents, suggested that families were dissatisfied with communications with doctors. The young people reported feeling that HCPs rarely sought their opinions and favoured direct communication with parents instead (Carter 2002). However, parents reported feeling that their interpretations of their child’s pain were dismissed by HCPs and that HCPs had labelled the family as dysfunctional or difficult because no organic explanation for reported symptoms could be found (Carter 2002). It is not clear whether this is a common experience due to the small sample size and lack of comparable research in this area. Professionals also articulated some concerns about their ability to interact effectively with families. In one UK qualitative study, HCPs reported feeling uncertain how to initiate discussions about psychosomatic disorders and involve psychological services without upsetting children, young people and their parents (Furness et al. 2009). Lindley et al. (2005) drawing on a retrospective analysis of 23 UK children with functional abdominal pain (FAP) suggest that parents of children with MUS may react negatively to HCPs’ opinions and investigative procedures by behaving in a confrontational manner, refusing to engage in psychological services, seeking second opinions and lodging complaints with hospital management. Lindley et al. (2005) reported that negative reactions to medical advice were associated with poorer outcomes for children and young people with FAP and contend that the potential for confrontation and © 2015 John Wiley & Sons Ltd

complaint can make it difficult for professionals to communicate effectively with families and manage symptoms appropriately. However, the study methodology and analysis strategy is poorly described and there appears to be a lack of quality research that has explored HCPs’ views on communicating with families of children with MUS. It is therefore difficult to assess whether the reported views are shared by HCPs working in other healthcare settings. Health beliefs Families reported strong beliefs in organic aetiology, wanted support from medical teams and were resistant to psychological intervention (Crushell et al. 2003, Guite et al. 2009, Jelbert et al. 2010, Hareide et al. 2011, Morris & Ogden 2012). In one small UK qualitative study of 13 mothers of children with recurrent unexplained symptoms, mothers reported feeling that symptoms were authentic and real, and required medical intervention (Morris & Ogden 2012). Two European qualitative studies reported that young people with CFS/ME attributed the condition to an undetected physical cause (Jelbert et al. 2010, Hareide et al. 2011), and a retrospective review of 138 young people with chronic pain shows that parents are more likely to associate high disability and pain scores with organic pathology (Guite et al. 2009). A survey of 28 parents of children with recurrent abdominal pain in Ireland found that the majority of parents of children with ongoing pain (n = 9 of 14) rated the input of a gastroenterologist as useful, but only four parents reported finding the involvement of a psychiatrist helpful, suggesting a reluctance to consider alternatives to the medical model of disease (Crushell et al. 2003). Quantitative and qualitative studies also suggest that children’s symptoms have a detrimental impact on family life, causing additional personal strain for mothers (Hunfeld et al. 2001, Morris & Ogden 2012), although no research has been conducted with fathers on this topic. The included studies suggest that HCPs may regard families’ health beliefs as a significant barrier to providing effective support to children and young people with MUS. Lindley et al. (2005) contend that families’ failure to consider psychological causes presents the most significant challenge to the successful management of MUS in children and young people, and risks compromising the child’s physical wellbeing. However, it is not clear how the presented data were interpreted to reach this conclusion. A retrospective quantitative study of young people with chronic pain showed that parental worries about 21

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young people’s health were associated with what the researchers termed ‘pain promoting behaviours’ (Guite et al. 2009). The researchers used this term to refer to parental actions that they believed inadvertently encouraged young people to report pain, such as parents deliberately reducing or altering the young people’s involvement in daily activities to accommodate their symptoms. Guite et al. (2009) argue that although parents’ actions may appear to be of benefit in the short term, they may affect the young person’s ability to effectively manage their pain and cope with sources of stress over the longer term. However, this study focuses on the retrospective review of parental responses to four questions that assessed their beliefs and worries about their child’s health and it is not clear how parental behaviours are categorised as ‘pain promoting’, why parents engage in these behaviours or the rationale for interpreting them as problematic. Healthcare settings Finally, families and HCPs appeared to perceive the setting of healthcare encounters to be an important factor in the delivery of support to children and young people with MUS. There is some limited evidence to suggest that families’ perception of the setting in which support is delivered may influence their willingness to discuss MUS with HCPs. A qualitative study of 16 young people with CFS and their parents in the UK revealed that families perceived the delivery of specialist therapy within ‘Psychological Medicine’ to be problematic, with families reporting feeling that professionals had wrongly interpreted symptoms as a mental health issue rather than a physical health condition (Dennison et al. 2010). However, HCPs working on paediatric wards in a UK hospital questioned the suitability of this environment for providing appropriate care and support to children and young people with MUS, suggesting staff lacked the time, resources and expertise to deliver intensive and tailored therapy during the course of their day-to-day work (Furness et al. 2009). However, it is not possible to infer whether this is an issue for other families and HCPs supporting children and young people with MUS or specific to the two UK sites studied due to the lack of research on this topic.

Discussion HCPs frequently encounter children and young people reporting MUS (Zuckerman et al. 1987, Hyams et al. 1996, Egger et al. 1999, Domenech-Llaberia et al. 2004, Eminson 2007, Geist et al. 2008, Furness et al. 22

2009, Carson et al. 2011). This group is at increased risk of co-morbid psychopathology and functional impairment in daily activities (Taylor & Garralda 2003, Konijnenberg et al. 2006, Crawley et al. 2009, Knook et al. 2011, Bould et al. 2013) and they require timely access to appropriate care and support to manage symptoms effectively. However, this review has identified a lack of well-conducted research that has examined families’ and HCPs’ perceptions of current services. Data were mainly collected from small homogeneous samples at single sites and data analysis procedures were often poorly described. Consequently, it is unclear if services are meeting families’ support needs and there appears to be insufficient research to inform the organisation and delivery of high-quality, evidence-based services for children and young people with MUS. This suggests that children and young people with MUS are at risk of receiving suboptimal care and support. The review has identified gaps in the literature and highlighted key areas for research. The included papers report cross-sectional studies that focus on small homogeneous groups of young women, mothers and doctors at single centres. The views of children, young men, fathers and other ethnic groups are under-represented. To our knowledge, there has only been one small mixed-methods study that has explored the perceptions of HCPs. At present, it is not clear whether the findings of the included studies can be extrapolated to families’ and professionals’ perceptions of other health and social care settings. As most papers report small-scale studies conducted in Europe, it is also not clear how findings compare intra- and internationally. Finally, most studies focus on CFS/ME and there is comparably less research into broader interpretations of MUS. The perception of patients with named syndromes may differ from the perceptions of patients with unclassified symptoms because a label may help them to access specialist services (Nettleton 2006). Future research needs to address the support needs of children and young people with MUS; families’ and professionals’ perceptions of current service provision; and how the organisation and delivery of health and social care services may be improved. There appears to be scope to develop welldesigned qualitative studies to explore families’ and professionals’ perceptions in greater depth and robust quantitative studies that use validated instruments to explore larger samples of children, parents and HCPs. This, in turn, will help to create an evidence base to inform high-quality practice that is acceptable to families and meets the complex needs of children and young people with MUS. © 2015 John Wiley & Sons Ltd

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Notwithstanding the limitations, it appears that families and professionals are ambivalent about the services provided to children and young people with MUS. The review highlights an apparent disjuncture between families’ and HCPs’ beliefs regarding the nature and best management of MUS. On the one hand, young people and parents report dissatisfaction with some HCPs’ approach to managing symptoms and feel there is a discrepancy between their health beliefs and HCPs’ actions (specifically the involvement of psychologists) (Carter 2002, Jelbert et al. 2010, Webb et al. 2011, Beasant et al. 2014). On the other hand, HCPs report feeling a lack of confidence in their capacity to support families due to limited time and expertise (Furness et al. 2009) and express concerns about families’ reactions to involving psychological services (Lindley et al. 2005, Furness et al. 2009). From the available evidence, communication between families and professionals appears to be problematic. Young people report experiencing negative reactions to reported symptoms and parents appear to be wary of HCPs focusing investigations on family practices rather than on children’s physical complaints (Carter 2002, Dennison et al. 2010, Jelbert et al. 2010, Hareide et al. 2011, Webb et al. 2011, Morris & Ogden 2012). Poor communication may lead to misunderstandings between families and HCPs resulting in young peoples’ and parents’ dissatisfaction with services. This, in turn, may encourage families to seek second opinions, request further medical investigations and refuse to engage with psychological services, leading to avoidable delays in appropriate management and treatment of symptoms (Lindley et al. 2005). Delivering services in psychological settings may also engender the belief that HCPs have misunderstood or do not believe young peoples’ reported symptoms (Dennison et al. 2010). Without clear evidence, it is likely that HCPs will engage in a process of ‘trial and error’ to establish the most successful strategies for working with families (Kozlowska et al. 2013). However, this may lead to inconsistencies in the care and support offered to children and young people with MUS that, in turn, may adversely affect the ongoing management of symptoms resulting in poorer health outcomes for young people. Specialists in the field of psychology and psychiatry advocate a family-centred approach to managing MUS in children and young people. HCPs are advised to acknowledge the existence of reported symptoms and recognise the adverse impact of symptoms on families’ lives (Leary 2003), listen to parents’ prior experience of the medical system (Kozlowska et al. 2013) and encourage families to engage in deci© 2015 John Wiley & Sons Ltd

sion-making (Kozlowska et al. 2012) to develop a medical consensus that is acceptable to all parties (Calvert & Jureidini 2003). However, it is not clear how widely these guidelines are communicated to other HCPs, particularly those working outside psychiatry and psychological services, and how easy they are to put into practice in different healthcare settings without suitable training and guidance. The review suggests that although children’s and young people’s symptoms cannot be explained medically, this does not mean that they are any less real to young people and their families (Morris & Ogden 2012). Furthermore, despite the lack of evidence, there is always the possibility that an organic cause has not been detected because the correct investigations have not been carried out (Eminson 2007). There is evidence to show that children with rare and/or serious health conditions have experienced significant delays in receiving a diagnosis because HCPs have dismissed non-specific symptoms as common childhood complaints without thorough and specialist investigation (Arksey & Sloper 1999, Dixon-Woods et al. 2001, Kharrazi & Kharrazi 2005). Dixon-Woods et al. (2001) contend that parents are an important source of information about their child’s health, but their expertise may be overlooked by HCPs who require objective and quantifiable evidence of disease. However, it is notable that the limitations of the biomedical model of health and the organisation of healthcare services are not addressed in any of the included studies. In contrast, it appears that the health beliefs and practices of families are subject to scrutiny and framed as inherently problematic in the management of MUS in children and young people (Crushell et al. 2003, Lindley et al. 2005, Guite et al. 2009). Finally, the findings of this review are comparable to research conducted with adults with MUS that emphasises patients’ dissatisfaction with services and HCPs’ uncertainties about supporting this patient group (Hartz et al. 2000). In addition, this review highlights the further complexities of supporting children and young people with MUS due to the involvement of parents in interactions with HCPs. Current evidence suggests parents may be the main source of information about symptoms, but may struggle to articulate invisible symptoms they are not directly experiencing (Webb et al. 2011). There is some limited evidence to suggest that HCPs would like to receive further information and support from CAMHS to help them provide appropriate individualised support to families of children experiencing MUS (Glazebrook et al. 2009), although further research is required to ascertain whether this view is 23

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shared by professionals working in other health and social care settings.

Conclusion Families and HCPs offer important insights into the support needs of children and young people with MUS and the availability and appropriateness of current services. To date, there has been limited research that has explored families’ and HCPs’ perceptions of healthcare services for children and young people with MUS. The available evidence highlights the potential for misunderstandings between families and HCPs regarding the underlying cause and appropriate management of reported symptoms. Families appear to have unmet support needs relating to information delivery, child–parent–provider communication, specialist care provision and healthcare settings. HCPs appear to require access to appropriate training and support to care for this group of families. The development of evidencebased clinical guidelines for children and young people with MUS, similar to those available for CFS/ME (NICE 2007), may be useful to inform the organisation and delivery of services that are acceptable to families. Future research needs to explore the complex support needs of families with a child with MUS, examine the training and support needs of HCPs, and identify the most appropriate way to organise care and deliver multidisciplinary services. Failing to provide appropriate and evidence-based services to children and young people with MUS may result in poor quality of life and higher healthcare costs in the long term due to extensive testing, repeat referrals and ineffective therapies.

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