Why do young people with CFS/ME feel anxious? A qualitative study

Abstract

Young people with chronic fatigue syndrome or myalagic encephalopathy (CFS/ME) (CFS/ME) experience higher levels of psychological distress than healthy controls and young people with other chronic illnesses, and it was recently demonstrated that 38% of this population scored above the clinical cut-off on the Spence Child Anxiety Scale. Subscales of social and separation anxiety were consistently high across gender and age groups. In this study, we used qualitative methods to help us understand more about these two types of anxiety in young people with CFS/ME. Eleven young people (age 12–18) were interviewed. Interviews were self-directed by the participants and were wide ranging. The transcripts were analysed using interpretative phenomenological analysis. Five superordinate themes were identified: social loss and adjustment; introduction of uncertainty and unpredictability; the vulnerable self; individual differences; and contributions towards recovery. Many themes were identical to those described in young people coping with other chronic illnesses in adolescence. In addition, young people with CFS/ME describe experiences associated with the perceived illegitimacy of this condition, namely: feeling unable to explain their illness; bullying from peers; disbelief; and distrust from adults around them. This becomes an additional challenge for these young people. Clinicians need to be aware of these problems, and offer appropriate support.

Keywords

Adolescents anxiety chronic fatigue syndrome ME qualitative

Introduction

Chronic fatigue syndrome or myalagic encephalopathy (CFS/ME) is defined as ‘generalised fatigue persisting after routine tests and investigations have failed to identify an obvious underlying cause’ (Royal College of Paediatricians and Child Health (RCPCH), 2004, p.12). The National Institute for Health and Clinical Excellence (NICE) guidance specifies that the fatigue must be persistent or recurrent and substantially reduce activity for more than three months (NICE, 2005; RCPCH, 2004) with one additional symptom from a list of a possible ten (headache, joint and muscle pain, sleep disturbance, cognitive dysfunction, painful lymph nodes, sore throat, flu-like symptoms, dizziness, nausea or heart palpitations) (NICE, 2005). CFS/ME in children under 18 is relatively common with a prevalence of between 0.06% and 2% depending on the definition and methodology used (Chalder, Goodman, Wesseley, Hotopf, & Meltzer, 2003; Farmer, Fowler, Scourfield, & Thapar, 2004; Jones, Nisenbaum, Solomon Reyes, & Reeves, 2004; Jordan et al., 2000, 2006; Rimes et al., 2007).

The relationship between CFS/ME and psychological difficulties is controversial, and polarised by psychiatric and physical causation models. Nevertheless, studies examining the levels of psychological difficulties in young people with CFS/ME consistently describe high levels of psychological difficulties compared to healthy controls and to young people with other chronic illnesses (Carter et al., 1999; Crawley, Hunt, & Stallard, 2009; Garralda & Rangel, 2004; Garralda, Rangel, Levin, Roberts, & Ukoumunne, 1999; Gray et al., 2001; van Middendorp, Geenen, Kuis, Heijnen, & Sinnema, 2001; Rangel, Garralda, Hall, & Woodham, 2003; Rangel, Garralda, Levin, & Roberts, 2000; Richards, Turk, & White, 2005; Smith, Martin-Hertz, Womack, & Marsigan, 2003). However, apart from one study (Richards et al., 2005) all have recruited from tertiary paediatric and psychiatric services, which might mean they represent the most complex patients. In addition, cross-sectional studies are unable to answer whether the psychological difficulties are causative or secondary. Neither does it help us understand the content and context of these young people’s psychological difficulties, over and beyond psychiatric labels.

Extrapolating from qualitative studies in adults with CFS/ME suggests that the illness and diagnosis of CFS/ME comes with additional psychosocial challenges perhaps not as frequently encountered when living with other more well understood physical illnesses. This is exacerbated by the debate over the name (CMO report, 2002). Experiences of stigma and a de-legitimisation of the person’s subjective experience seem common (Arroll & Senior, 2008; Dickson, Knussen, & Flowers, 2007; Ware, 1993). Other studies show the importance participants put on other people understanding CFS/ME as a physical problem. This has been postulated to help avoid feelings of guilt, stigma or blame related to the illegitimacy of the illness (Guise, Widdicombe, & McKinlay, 2007). However, while this has been shown in an adult population, Richards (2004) suggests the impact of CFS/ME is likely to be different for young people. In particular, a long period of illness and absences from school, friends, and activities can have a larger impact on academic, social and emotional development due to the rapid changes taking place during this developmental stage (Boice, 1998).

To date we found one qualitative study which investigated the experiences of young people with CFS/ME in general (Jelbert, Stedmon, & Stephens, 2010). This study described five themes which echoed many of the psychosocial challenges described in the adult qualitative literature: seeking understanding; stories of loss; the difficult emotional experience; influences on the illness; and how I am now.

Simultaneously, a previous questionnaire study had identified particularly high levels of separation anxiety and social phobia in children and young people with CFS/ME (Crawley et al., 2009). Following on from this finding, we wanted to understand more about the development, content and context of these types of worries in young people with CFS/ME as seen from their own perspective. We therefore went on to interview 11 young people with CFS/ME and high levels of separation and social anxiety about their psychosocial experiences of living with CFS/ME and high levels of anxiety. We analysed the data using Interpretative Phenomenological Analysis (IPA) (Smith, Flowers, & Larkin, 2009).

Method

Participants

We recruited young people aged between 12 and 18 years, with a clinical diagnosis of CFS/ME, who scored above the 90^th percentile cut off on the social phobia and/or separation anxiety subscales of the Spence Children’s Anxiety Scale (SCAS) (Muris, Schmidt, & Merckelbach, 2000). Young people were recruited from the outpatient clinic during their assessment. We therefore excluded patients who were too unwell to attend hospital.

Procedure

The assessment clinician asked for verbal consent from the young person and their parents for a researcher to send an information sheet and consent form. Participants who returned signed consent forms were contacted by phone to arrange the interview. The participants could have the interview at home or in the clinic. Interviews were conducted for a maximum of 40 minutes with an optional break in the middle. No single session was longer than 20 minutes unless the child was able to manage significantly longer in a single session in school/college. Prior to the interview, young people were given a topic guide and were able to opt out of areas they did not want to discuss. The topic guide was designed on the basis of a detailed literature review as well as feedback from experienced clinicians, research supervisors and Association of Young People with ME (AYME). The guide was designed to specifically investigate the young people’s experience of social phobia and separation anxiety and covered the areas of family life/separation anxiety, school and friends/social anxiety, as well as changes to identity. The topic guide (see appendix) was used as a guide only, and topics were dealt with as they arose in the participants’ stories. The interviews were self-directed by the participants and were wide ranging. All interviews were recorded on a digital recorder and transcribed verbatim by the researcher.

Measures

Prior to clinical assessment, inventories measuring fatigue, physical function, anxiety/depression, number of symptoms, school attendance, and pain are routinely collected. Fatigue is measured using the 11-item Chalder Fatigue Scale which is scored using the 0–3 method for scoring each question (0 for ‘less than usual’, 1 ‘no more than usual’, 2 for ‘more than usual’ and 3 for ‘much more than usual’) (Chalder et al., 1993). Physical function is measured using the 10-item physical function subscale of the short form (36) health survey (SF-36) (Garratt, Ruta, Abdalla, Buckingham, & Russell, 1993). This is scored between 1 (‘yes limited a lot’) and 3 (‘no not limited at all’) for each question so that children with the worst physical function scored 10 while those with good physical function scored 30. The Hospital Anxiety and Depression Scale (HADS) is used to screen children aged 14 years and older for low mood and anxiety symptoms (Snaith, 2003). Anxiety is measured using the Spence Child Anxiety Scale (SCAS) (Spence, 1998; Spence, Barrett, & Turner, 2003). It was developed to correspond to the DSM-IV diagnostic categories for anxiety in children and consists of 38 items divided into six subscales: physical injury fears; obsessive-compulsive disorder; panic attacks; separation anxiety; social phobia and generalised anxiety disorder. The items are scored on a four-point scale; never, sometimes, often or always. Normative data for this scale have been derived for children aged 12–18 years (Muris et al., 2000). School attendance is collected as a single item inventory as the percentage of possible time at school (0 = 0%, and 7 =100% attendance). Number of symptoms is recorded using a symptom list derived from the Centre for Disease Control criteria (Fukuda et al., 1994) and the RCPCH guidelines (RCPCH, 2004). A visual analogue scale is used to measure pain, with a score of 0 for ‘no pain’ and 100 for ‘pain as bad as possible.’

Statistics

To check that those interviewed were comparable to the whole cohort assessed in the out-patient clinic, we compared those interviewed with those not interviewed using standard two-tailed T tests on the measures of age, fatigue, SF-36, pain, number of symptoms, HADS, SCAS, school attendance and time to assessment in months. Chi-square was used to compare the gender distribution.

Qualitative analysis

The analysis was conducted according to guidelines for IPA by Smith et al. (2009). IPA is increasingly popular in qualitative health psychology research, and is particularly suited to exploring the psychological impact of health problems as it aims to identify how people make sense out of their world (Smith et al., 2009).

The interviews were transcribed by the researcher, and the transcripts were read a number of times, noticing initial thoughts. This was done for each transcript individually. Then the researcher used the second column to note down provisional themes. The identified themes for each individual transcript were then listed. Themes from all transcripts were subsequently combined and organised as connections and similarities emerged across the interviews. At this point, a narrative account of each identified higher order theme and its sub-themes was detailed in the research log to facilitate a deeper understanding. The themes were cross-validated by an external researcher. During these stages the researcher regularly met with a second independent researcher to check interpretation of the themes, and their support in the data. In order to enhance the reliability and validity of the analysis, a data audit trail was kept. Finally, quotes illustrating each theme were selected. It was also ensured that all participants were represented in the write-up, which demonstrates that no cases were excluded from the analysis (Meyrick, 2006). Pseudonyms were used for all participants.

Ethics

This study was scrutinised by an ethical committee in the south west of England and given a favourable opinion: REC number 08/H0106/141.

Results

Twenty-two eligible participants were identified in the clinic between March and October 2009. Of these, 10 did not return consent forms, one did not want to take part and 11 consented to the study, of whom 10 were interviewed at home and one in the clinic. The participants’ ages ranged from 12 to 18 years, with a mean of 15 years. Nine were girls. Table 1 shows that there was no statistically significant difference between the group interviewed and the cohort apart from on the SCAS which was significantly higher for the sample (mean 41.8/29.6, CI = 1.4 – 23, N =570). Table 1 further shows that the 10 young people who were eligible for the study, but did not return the consent forms, were no different to the interviewed young people apart from on the measure of ‘time to assessment’ which was longer for the non-responders (mean 38.5/17.2, CI = 0.3 – 42.3, N = 22).

Table 1.

Statistical comparisons between the whole clinic cohort, study sample, and those who did not respond to the invitation to be part of the study (non-responders).

Characteristic	Whole cohort		Study sample		Mean difference	P value	Non-responders		Mean difference	P value
	N		N		(95% CI)		N		(95% CI)
Age: mean (SD)	746	14.3 (2.8)	11	14.6 (2.5)	0.7 (−1.0 to 2.4)	0.4	10	14.6 (2.0)	0.3 (−2.1 to 2.0)	0.9
Gender: proportion.	749	228/521 = ♂	11	2/11 = ♂	N/A	0.38	10	1/11 = ♂	N/A	1
Fatigue: mean (SD)	660	23.9 (5.6)	11	22.5 (8.6)	−1.4 (−4.8 to 2.0)	0.41	10	25.6 (4.5)	3.1 (−3.2 to 9.5)	0.3
SF-36: mean (SD)	642	20 (5.5)	11	21.9 (5.4)	1.7 (−1.6 to 4.9)	0.32	10	18.4 (5.1)	−3.5 (−8.3 to 1.3)	0.14
Pain: mean (SD)	598	46.6 (30.3)	10	44.9 (36.6)	−1.7 (−20.8 to 17.3)	0.86	9	50.6 (16.1)	5.7 (−22.3 to 33.6)	0.67
Number of symptoms: mean (SD)	692	8.2 (2.7)	9	8.3 (2.3)	0.1 (−1.7 to 1.9)	0.92	10	8.5 (1.4)	0.17 (−1.66 to 2.00)	0.85
HADS, anxiety: mean (SD)	497	8.6 (4.3)	8	9.9 (4.0)	1.3 (−1.8 to 4.3)	0.21	10	8.8 (3.7)	−1.1 (−4.9 to 2.8)	0.56
HADS, depression: mean (SD)	496	7.4 (3.7)	8	7.8 (4.5)	0.4 (−2.2 to 3.0)	0.77	10	6.3 (3.6)	−1.5 (−5.5 to 2.6)	0.46
School attendance: mean (SD)	722	3.9 (2.3)	11	4.9 (2.3)	1.1 (−0.3 to 2.4)	0.14	10	4.8 (1.9)	-0.1 (-2.0-1.8)	0.9
Months to assessment: mean (SD)	694	25.2 (25.1)	11	17.2 (13.7)	−8.0 (−22.9 to 6.9)	0.29	10	39.6 (29.3)	22.4 (1.9 to 42.9)	0.03
SCAS : Mean	570	30.0 (18.1)	11	41.8 (16.1)	12.2 (1.3−23.0)	<0.00	10	34.9 (9.6)	−6.9 (−19.2 to 5.3)	0.3
SCAS: subscales
- Social phobia (SD)	640	6.6 (4.1)	11	9.8 (4.3)	3.1 (0.7−6.0)	<0.00	10	9.1 (3.0)	−0.7 (−4.2 to 2.7)	0.67
- Separation anxiety (SD)	646	4.3 (3.9)	11	6.3 (4.0)	1.9 (0.3 to 4.3)	0.05	10	3.8 (2.0)	−2.5 (−5.4 to 0.5)	0.1
- Panic/agoraphobia (SD)	637	4.9 (4.6)	11	6.7 (4.0)	1.8 (−0.9 to 4.6)	0.1	10	5.0 (3.4)	−1.7 (−5.1 to 1.7)	0.3
- OCD (SD)	638	3.5 (3.5)	11	6.2 (3.2)	2.6 (0.2 to 5.1)	<0.00	10	3.9 (2.6)	−2.3 (−4.9 to 0.4)	0.09
- Generalised anxiety (SD)	645	6.3 (4.0)	11	9.0 (3.2)	2.6 (0.2 to 5.0)	0.02	10	8.0 (3.7)	−1.0 (−4.1 to 2.1)	0.51
- Physical injury fears (SD)	655	3.9 (2.8)	11	3.9 (2.4)	−0.4 (−1.7 to 1.6)	0.5	10	5.0 (2.7)	1.1 (−1.2 to 3.4)	0.33

HADS: Hospital Anxiety and Depression Scale; SCAS: Spence Children’s Anxiety Scale; OCD: Obsessive Compulsive Disorder.

Qualitative results

The qualitative analysis of the interviews produced five super-ordinate themes, which each had between two and four subordinate themes each (see Table 2). The themes are detailed in the text to provide a rich description of the breadth and complexity of the young people’s experiences of CFS/ME and anxiety.

Table 2.

Summary table of superordinate and sub-themes.

Superordinate themes:		Sub-themes:
Theme 1: social loss and	1	The loss of normal adolescent life.
Adjustment	2	Friendships were put to the test.
	3	Increased worries about school work.
	4	The need for adjustments to family relationships.
Theme 2: introduction of	1	Uncertainty about the future.
uncertainty and unpredictability	2	Uncertainty of the validity of CFS/ME: feeling disbelieved.
	3	Feeling uncertain about how to explain CFS/ME.
Theme 3: the vulnerable self	1	Identity confusion.
	2	Increased emotionality.
Theme 4: individual differences	1	Content of anxiety.
	2	Onset of anxiety.
Theme 5:contributions towards	1	Good relationships.
Recovery	2	Awareness of CFS/ME.
	3	Hope.

Theme 1: Social loss and adjustment

When asked ‘what is the worst thing about CFS/ME’, some young people talked about the internal physical experience of CFS/ME: ‘the utter exhaustion; it is really horrible’ (Vicky). However, most others emphasised the social implications of the illness: CFS/ME was perceived to be ‘socially crippling’ (Imogen). Due to the disabling fatigue, long periods were spent absent from life outside the family home. Four sub-themes were identified: the loss of normal adolescent life; friendships being put to the test; increased worries about school work; and the need for adjustments to family relationships. These sub-themes will be presented in turn.

The loss of normal adolescent life

The prolonged isolation due to the illness brought both loneliness and boredom: Ben was facing ‘missing my friends, not playing basketball. Just constant boredom’. Being a part of their peer group was considered important. Feeling stuck, left out and different from friends was painful and upsetting:

I remember at prom, I remember everyone else dancing and I was the only one sat down. And ohh…like… like… it was horrible. That feeling, that feeling of being sat down watching everyone else dancing, it’s kind of how I felt when I was in bed and everyone else was out. It felt like I was sort of watching everyone else… like watching like… everything revolving around me, and I could only sit there, or lie there. (Olivia)

Friendships were put to the test

The young people generally perceived their relationships to be put to the test following the onset of CFS/ME, both by their absence, and them continually having to turn down invitations. Some described their friends as unwilling to make necessary adjustments. They felt rejected and forgotten about, and there was a strong sense among the young people that after becoming ill, you ‘learn who your true mates are’ (Emily):

I asked her [best friend] to come up and she got all ‘Yeah, I will’. But then I found out that she had gone out with her boyfriend. She would like always stitch me up: Obviously it got more upsetting to me. And she was said, ‘I cannot deal with ill people’. I said ‘I am your mate I need you to be there for me’. ’Cause like none of my mates at all come and see me. None of them rang me to see how I was and that. (Miriam)

Half of the participants (6/11) had also experienced bullying from their classmates regarding their illness. This was related both to the nature of the illness and the uncertainty of the validity of CFS/ME:

They make like little jokes about it like; ‘O no, he cannot go and get his racquet… No that takes energy’… It’s not even funny. They have like little smirks on their faces. (Ben)

Like one of them says I am a ‘skiver’, but they only joke about it. Then one of the girls was really horrible on MSN. (Cara)

Her boyfriend had been saying ‘Hayley has Down Syndrome’, and all like that.

(Hayley)

Increased worries about school work

‘Worrying about school work’ was the most pervasive theme in this analysis. The young people missed large amounts of school and were unable to catch up on the work due to a restriction on their activity levels. While the young people varied in their commitment to school prior to getting ill with CFS/ME, being unable to attend school seemed to bring home its significance:

’Cause this last exam I just did I hadn’t been to at least half of the lessons on the subject. So I suppose it was more nerve-wracking than it had been before. (Imogen)

I am worried that I am going to return, and they are going to be so far ahead of me in term of topics and things that I haven’t learned. So I do kind of worry about that. (Sophie)

Worry about returning to school was related to anxiety about the potential of feeling over-stimulated in the school environment. More specifically, anxiety about how feeling fatigued might impact on their ability to socialise and concentrate in class made thinking about school-reintegration difficult:

I do kind of worry about going back to school and returning back to that whole sort of atmosphere. ’Cause I do find it hard to concentrate, with the noise at school. And then, I kind of, when I return, because everyone is so ‘buzzy’ and happy and, and I, it does sort of drain me. I find it hard to keep up with them ’cause they are all really energetic. (Sophie)

The need for adjustments to family relationships

Having CFS/ME often posed a challenge to the young person’s whole family. Some described increased closeness, and others increased conflicts with their parents and siblings. Whereas the young people had previously spent increasing amounts of time with friends, they now relied more on their family for both emotional and practical support. There was a perceived lack of age-appropriate independence, and some felt guilty and a burden on their family’s resources:

It’s quite hard because my mum is on unpaid leave, so we don’t have much money. So I feel a bit annoyed, ’cause my sisters had to stop swimming and piano ’cause it costs too much, and I feel a bit guilty for that, because they cannot learn that because of me. (Emily)

Theme 2: The introduction of uncertainty and unpredictability

The young people talked about CFS/ME increasing uncertainty and unpredictability in many areas of their lives including the future. Similarly, the uncertainty of the validity of CFS/ME as a diagnosis impacted on their relationships for most of the young people, as some teachers, friends and family became distrusting and invalidating of their personal experiences through not believing in their fatigue. The young people felt it was difficult to explain CFS/ME to their social circles, and this may have perpetuated the lack of understanding. These three sub-themes will be presented in turn.

Uncertainty about the future

The uncertainty of the trajectory of the illness often made young people question the plans they had made for the future, particularly their ability to perform in exams and get accepted to colleges or universities. The two boys in the study had aspirations of a career as professional sportsmen, but having CFS/ME made them doubt their ability to continue with their sports:

I wanted a career in basketball. But now it’s not looking too good, I’ve got like six months off now, and it’s looking like I’ll have like eight months off, until I am better, so it’s not looking very good. (Ben)

Uncertainty about the validity of CFS/ME: Feeling disbelieved

All the participants talked about being disbelieved about their fatigue. Suddenly, unpredictability was introduced into relationships, and some teachers, family or friends would seem to lack trust in them, and would dismiss their illness experiences as fabricated:

I just don’t think some of them [teachers] in particular were that supportive, and they didn’t really realise what was going on, and they thought I was just having days off, like faking ill really. (Vicky)

People like don’t understand it really, cause like my dad and all that, and my brother, they just thought I was being lazy, they didn’t understand it at first, they just thought I was being lazy. (Cara)

The young people talked about people attributing their inability to do school work, or attend activities, primarily to a lack of motivation or being lazy. Having their illness misinterpreted as laziness was difficult for young people, as the young people understood laziness to be a negative attribute:

I don’t want people to think I am not pulling my weight, or trying to cop out of things, or I cannot be bothered to do that, not putting as much effort in as them, just things like that. I didn’t want people to think less of me, especially my teachers, and friends.

(Olivia)

Feeling uncertain about how to explain CFS/ME

Being given a diagnosis of CFS/ME did not always help the young people to legitimise their illness experience with regards to their social networks. The lack of medical and outwardly visible physical signs of illness made it difficult to explain CFS/ME:

I don’t know what to say. And that worries me. People ask about it and I don’t know what to say. […] So I just say I got ME. And then they say, what is that? And I cannot explain it, ’cause I don’t really know myself, ’cause I didn’t really know until they told me. So I just sort of say: ‘Don’t worry, you won’t understand or…’ ’cause I don’t really understand. I don’t think anyone does, do they? (Miriam)

Many talked about how others would trivialise the fatigue associated with CFS/ME, and emphasise that tiredness is a common experience. Thus, it seems that the use of the word ‘tired’ lacked explanatory power, and few alternatives were available:

’Cause saying like you are tired all the time, it’s difficult for people to get really, ’cause you are just tired. Lots of people get tired. (Sophie)

Theme 3: The vulnerable self

The previous two themes describe experiences associated with social aspects of the young people’s lives. Theme 3 describes the internal, individual experience of CFS/ME and details two sub-themes: identify confusion and increases in emotionality. These two themes, and the importance of the meaning-making related to the specific symptoms of CFS/ME, will be presented in turn.

Identity confusion

In talking about themselves and their identities, the young people frequently drew on language relating to their hobbies or personality characteristics. However, as these areas had changed after the onset of CFS/ME, they often expressed confusion about their identity. Cara illustrated how her pre-illness self was someone who did what she perceived to be good activities: ‘I used to love walking and swimming and horse riding and that’. She contrasted this with what she perceived as less good current activities: ‘Now I am just like watching telly, and that is not very good’. Sophie further illustrates how a perceived change in activity levels due to the symptoms of CFS/ME led to her questioning her sense of self:

’Cause when I am well, I am really energetic and sporty, and outgoing, but at the moment I feel as if I have changed, because I’ve not been as well, I feel like I have changed as a person, and I am not as energetic and outgoing and stuff… which has been quite hard to get my head around, ’cause I don’t really understand what I have kind of turned into… (Sophie)

Sophie talks about her change in activity levels and personality not as an unavoidable and temporary impact of the CFS/ME, but she questions whether this is reflecting a change in who she is, a change in her ‘Self’.

The symptoms of CFS/ME, such as weakness, tiredness, sensitivity to sound and light, brought an increased sense of fragility and vulnerability to the young people. For some it led to worries about travelling, germs, answering the door, talking on the phone and self image. For most, feeling fragile and vulnerable also lowered their self-confidence, and this seemed to be a particularly difficult change for those who had felt socially competent before:

I just don’t think I am as confident as I used to be in that I used to just be able to talk, but now I find social situations really difficult unless it is like me and just one other person. And if it is like a group of us, I just stick to one person that is close to me. They’ll know what is going on, but I used to be really good in those situations and things, and that was a bit hard. (Vicky)

Increased emotionality

The young people also talked about feeling increasingly irritable, sad and worried following the onset of the illness:

It’s been lots since been ill, it’s been lots of things happening that has made me get really emotional. And sort of it seems like it’s affected me more than what it would have, if it had happened before I got ill. (Hayley)

Imogen further describes how being a teenager, and knowing about the emotional changes this commonly brings, complicates her understanding of the emotional impact of having CFS/ME:

’Cause I have had ME for so long, I don’t know what I am like without it. I haven’t been a teenager without it. I was a kid before I got it, I would have changed anyway. I am more grown up. I wonder, ’cause I get ehmm… Tired and grumpy and stuff, and … Would I be like that if it wasn’t for ME? ’Cause I know, I do know for a fact how tired I am now, but how would I be if I was… ’cause I was only like 12… and a lot has happened since then. (Imogen)

The increase in feelings of sadness, irritability, worries and tearfulness was generally perceived as unwelcome, and a further burden on both themselves and their surroundings: ‘It is always bad having a negative friend rather than a positive friend’ (Vicky). Some had friends and family that they could talk to, but not everyone had someone that would listen to them. Some felt that friends did not want to hear about their illness all the time: ‘[it] sort of puts a downer on the day’ (Sophie).

Theme 4: Individual differences

While there were similarities among the young people in this study, it was clear that the participants differed significantly from each other in the content of their worries and the perceived onset of their anxiety.

Content of anxiety

Although a high level of anxiety was pervasive among the participants, the content of their anxieties varied. Two participants referred to being a perfectionist, e.g.: ‘I think I’m a bit of a perfectionist’ (Miriam), while the remaining nine did not talk specifically about this. Three other young people described high levels of separation anxiety. For some this seemed linked to long-standing night time worries and sleeping difficulties:

At night time there are a lot of worries about the house being broken into and stuff. […] Just other random things like that… that the house is on fire; that I will have to put on my clothes and go outside. If the house gets broken into, that my mum and dad won’t hear me. (Louise)

Onset of anxiety

Some of the young people recognised that the onset of their anxieties was quite some time before the onset of the CFS/ME. This was the case for the young people described in the above paragraph; the two young people who used perfectionism as a strategy to cope with anxiety and the three young people with particularly high levels of separation anxiety. For others the onset was felt to be after the onset of their condition.

As an example, nine of the young people talked about not having been particularly into school nor excessively concerned about school work prior to CFS/ME:

I don’t think I have been a really good person to do work; I have always been the person to mess around in lessons and stuff. (Ben)

These nine then became worried about school work after the long absence from school, and with the increasing sense of loss of control over their school work and future. Similarly, two participants talked about separation anxiety becoming a problem when they were isolated at home with CFS/ME and felt ill and vulnerable.

Theme 5: Contributions towards recovery

Within the stories of difficult experiences, the young people also stressed what had been helpful during their recovery: supportive relationships; information about CFS/ME being passed on to school and friends; and finding hope for recovery. These themes will be presented in turn.

Good relationships

Some of these young people’s friends, family and teachers adapted positively to the challenges created by their illness. This included practical help, such as giving lifts or coming around for frequent, short visits. The participants also talked about feeling emotionally supported, helped, understood and considered. The significance of good relationships for recovery is powerfully illustrated by Hayley:

It was really hard until four months ago when I didn’t have Matt [boyfriend] and now I am so happy to have him here. And it’s lucky. And after losing my mates, if I didn’t have him, I don’t know what I would have done. I’d just feel useless, wouldn’t want to do anything, wouldn’t know what to do, just stay in my room all the time, just not do anything. (Hayley)

Awareness of CFS/ME

When their social/support network had been informed about CFS/ME, some of the young people felt that others understood that CFS/ME was ‘a serious illness affecting your life in a big way’ (Sophie). This seemed to help remove the feelings of distrust. Sophie even felt that her recovery had depended on the information her school had been giving to her friends:

I think that informing people around, the more people know about it, the better they are going to be in dealing with it. If the school hadn’t been telling all my friends, I don’t think I would be where I am now recovering. So I think that is going to be one of the main things, it needs to be talked about. And let people know what it is, so they are aware of it. (Sophie)

Hope

The young people talked about the importance of having hope for recovery. Before being given a diagnosis they were in a state of uncertainty, not knowing if there was anything wrong, or what could be done. Ben described how important it had been for him to be given a name for his illness and a plan for recovery:

When it first happened, I felt sort of like lost. I didn’t really feel myself, but then after [the hospital appointment], after knowing what I had, I had like a plan to get through it, and I started feeling more like myself. (Ben)

Discussion

The young people in this study described pervasive losses associated with having a chronic illness in adolescence as well as complications of being diagnosed with an illness that is poorly understood and stigmatising. The majority of the young people interviewed related their worries to difficult experiences following the onset of the CFS/ME, such as losing out on normal teenage life, conflicts with friends, worries about school and the future, feeling vulnerable, confused about their identity, disbelieved and distrusted about the validity of their illness. In contrast, the stories about separation anxiety in the young people in this study appeared more diverse. Some talked about feeling more anxious about separation from their parents following the onset of the illness. However, a minority (3/11) reported significant separation anxiety also prior to being given a diagnosis of CFS/ME.

Strengths

This is the first study to use qualitative methods to investigate the development and experience of anxiety in young people with high levels of social phobia and separation anxiety CFS/ME. It describes in detail both the content and context of their worries. A further strength of the study is the open interview technique which allowed the young people to talk about what concerned them most. This means that results inevitably represent a broader range of experiences than stories about social and separation anxiety. For example, the stories included other difficult emotional experiences such as sadness, guilt and shame. What links these experiences to our focus on anxiety is how over time they contribute to a general sense of uncertainty, unpredictability and worries about the self, others and the future; the cognitive triad of anxiety (Blackburn & Davidson, 1990). We were also able to show that, as we recruited young people specifically on the basis of high levels of anxiety, the participant group scored significantly higher on a questionnaire measure of anxiety (the SCAS) compared to the general population of young people with CFS/ME accessing a specialist CFS/ME service. On all other screening measures they were representative of the clinic population (age, gender, levels of fatigue, pain, school absence, physical functioning, number of symptoms and depression). The difference between those interviewed and the clinic population was much higher for the SCAS then for the HADS. This may be because we selected children on the basis of the SCAS, because there were more items in the SCAS (44, compared to nine on the HADS) or because the HADS is not a useful screening tool in younger children with CFS/ME.

Limitations of this study

Similar to most other studies in this population, the young people were recruited from a specialist service and it is likely that our participants represent the most complex end of the spectrum of young people with CFS/ME. We also recruited young people with the highest anxiety scores; thus our findings are not necessarily generalisable to all young people with CFS/ME.

We had a relatively high level of non-response, and non-responders were more likely to have been unwell for longer. It is possible that this group had more difficulty accessing specialist help. It is also possible that their presentation was more complex and they felt unable to talk about their experiences.

Because children were fatigued, we limited interviews to 40 minutes and were therefore unable to ask about other thoughts and feelings such as anger or low mood. This limits our ability to interpret these findings and should be the subject for future research.

Comparisons with other literature

Our results are consistent with themes described in a smaller IPA study including young people with CFS/ME (Jelbert et al., 2010). That study additionally described stories of personal growth. This may be due to their participants having recovered, whereas the young people in our study were recruited shortly after assessment.

Our results are also consistent with the adult literature, which describes an identity crisis and sense of loss following the onset of CFS/ME (i.e. Charmaz, 1983; Dickson, Knussen, & Flowers, 2008). Additionally, the teenagers in our study talked about the interaction between the challenges of identity development in adolescence and the loss of valued roles and personality characteristics due to long term illness. Similar processes have been highlighted both in other studies and theoretical literature (i.e. Boice, 1998; Eccleston, Wastell, Crombez, & Jordan, 2008; Meijer, Sinnema, Bijstra, Mellenbergh, & Wolters, 2000; Roche & Tucker, 2003).

Meaning of this study

A range of studies have shown that most chronic illnesses in childhood entail significant changes, experience of loss and need for adjustments (theme 1) which often lead to psychological distress (i.e. Suris, Parera, & Puig, 1996; Woodgate, 1998; Woodgate, Degner, & Yanofsky, 2003). Indeed, an increase in emotionality was described by the young people in this study (theme 3).

However, our study suggests that an illness such as CFS/ME, which is less well known and understood, comes with additional psychological challenges for the young people (theme 2). In line with the adult qualitative literature (Arroll & Senior, 2008; Dickson et al., 2007; Ware, 1993) the perceived illegitimacy of CFS/ME as an illness was an important theme in these interviews. This was seen in three areas.

Primarily, the young people described being bullied by peers. They talked about name-calling and had their absence due to illness dismissed as deliberate ‘skiving’. Both direct and indirect bullying in childhood is strongly associated with a range of poor psychosocial outcomes (i.e. van der Wal, de Wit, & Hirasing, 2003).

Secondly, many of the young people felt that the name of the illness did not fit their overwhelming experience of complete physical and mental exhaustion. ‘Fatigue’ was felt to be too close to the ‘normal tiredness’ experienced by most normal human beings on a day-to-day basis. It was important for the young people that their social circles understood that their experience was much more severe than this tiredness. This can be understood within the strong narrative in the western world that it is weak or lazy to ‘give in’ to normal tiredness; for example embedded in the ‘protestant work ethic’ (Greenberg & Furnham, 1990; Rotenberg, 2003). Within this belief system, serious physical illness is one of the few valid reasons for not working hard. The young people felt that the name ‘Chronic Fatigue Syndrome’ did not denote a serious illness. Thus, it was not always seen as sufficient to legitimise their absence, lowered activity levels, and special treatment to their social networks. Most of the young people had been called lazy by significant others. For some, a diagnosis of CFS/ME could not fully remove the feelings of shame they associated with lowered activity levels. Feelings of being shamed are related to high levels of psychosocial distress and have been hypothesised to be a trigger of depression (i.e. Gilbert & Andrews, 1998).

Finally, linked to this, the young people frequently talked about being anxious and distressed about experiences of distrust and disbelief from family, teachers and friends following the onset of the CFS/ME. All of the young people had had experiences where they were accused of pretending to be ill, or exaggerating their experience of symptoms. It can be argued that what these young people are describing is an example of what Linehan (1993) calls an ‘invalidating environment’; the young person’s internal world is dismissed in favour of the story of more powerful others. In writing about ‘invalidating environments’ Linehan (1993) explains that ‘the expression of private experiences is not validated; instead it is often punished and/or trivialized […]. The individual’s interpretations of her own behaviour, including the experience of the intents and motivations of the behaviour, are dismissed’ (chapter 3, pp. 66–96).

Together, these experiences seem to shake the young people’s beliefs and assumptions about others and the future, which have become uncertain and unpredictable. Having their beliefs challenged in this way was emotionally difficult, and came at a time when the young people already experienced themselves as being fragile and vulnerable. The experience of anxiety has been conceptualised as: the unpredictability of the future; the dangerousness of others; and the vulnerability of the self (Blackburn & Davidson, 1990). The young people’s stories illustrate meanings behind anxiety: for some the illness brought a sense of personal vulnerability and unpredictability of the future; for others the perceived rejection of the validity of their experiences of CFS/ME led to fear and unpredictability.

In summary, children with CFS/ME appear to experience the ‘normal’ loss and need for adjustment associated with a chronic illness. This is made worse by the introduction of uncertainty and unpredictability experienced in children with CFS/ME because treatment outcomes, uncertainty about the future and the validity of the illness are not clear. In the context of a vulnerable self this would commonly lead to anxiety. However, how the anxiety is expressed is different for different children. It is likely that the psychological impact would depend on a range of circumstances, for example the young person’s previous parenting experiences.

Whilst some children describe factors that contribute to recovery (good relationships, awareness of CFS/ME and hope), it is likely that for some the behavioural responses induced by their anxiety will reduce the chance for recovery (Deary, Chalder, & Sharpe, 2007), for example by pushing oneself beyond one’s current physical stamina, over-resting or not going out due to the fear that one may be seen walking around appearing physically ‘normal’.

Implications for clinical practice

While the uncertainty and unpredictability associated with CFS/ME and the problems children experienced with the label ‘CFS/ME’ were important themes in this study, some also talked about how getting a diagnosis and plan for rehabilitation helped to remove uncertainty, and provide hope for recovery.

The experiences of stigma and disbelief highlighted that there may be a need for working with the systems around the child, for example families and schools, to increase understanding about CFS/ME and enabling them to support children undergoing a rehabilitation programme.

Following the onset of illness, our participants reported lowered self-confidence and increases in negative emotions associated with the many losses they had expereinced. A reaction of increased emotional distress is normal following the onset of chronic health conditions (Suris et al., 1996; Westbrook & Viney, 1982), and normalising psychological distress has been hypothesised as being helpful in the adjustment process (Moorey, 1996). This might help young people with CFS/ME to understand that increases in emotionality do not reflect a change to core aspects of their identity, but is a response to these major changes to their life circumstances.

Furthermore, this study indicates that young people may be ambivalent about a diagnosis of CFS/ME, and this could limit their desire to engage with a treatment programme. For the young people that do not engage with a rehabilitation approach, it is possible that a therapeutic approach such as Motivational Interviewing (MI) may be helpful. While MI has a growing evidence base in adolescents, we have not found any papers evaluating this approach in CFS/ME to date (Chung, Burke, & Goodman, 2010; Erickson, Gerstle, & Feldstein, 2005).

Finally, we also show that anxiety is not the same for every young person with CFS/ME either in content (only some appeared to be perfectionist) or in onset (before or after the onset of CFS/ME). It seems likely that young people with CFS/ME and high levels of anxiety may require individualised treatments, tailored to the different types of anxiety as part of a rehabilitation programme for CFS/ME.

Conclusion

The young people in this study described the complex psychosocial implications of living with a chronic illness in adolescence and highlighted the specific challenges of living with an illness such as CFS/ME. The young people interviewed continue to experience a lack of awareness and acceptance of CFS/ME, which seems to put an increased burden on these young people in addition to the already major social and personal impact of chronic illness in adolescence. This study suggests that the high levels of anxiety shown in young people with CFS/ME may, in the least, be intensified by both the psychosocial impact of the illness as well as the uncertainty of the diagnosis of CFS/ME.

Footnotes

Appendix: Topic guide

Relevant questions from the SCAS:

Researcher: when you talk about these things, I’ll try to help you talk about it so I will ask more questions like:

Supplementary questions (example):

Can you tell me about a time that happened?

When did that first become a problem for you?

Do you remember worrying about this before you had CFS/ME?

Why do you think this is a problem for you?

Do you feel it is getting worse or better lately?

Acknowledgements

We are grateful to all the young people who took part in this study and to the clinicians from the service who helped recruit them. Thanks to Dr Simon Collin who helped with the data analyses.

Funding

This work is produced by EC under the terms of a Clinician Scientist award issued by the NIHR. The views expressed in this publication are those of the author(s) and not necessarily those of the NHS, the National Institute for Health Research or the Department of Health.

Author biographies

Hanne Fisher is a Clinical Psychologist and completed this study during her training at the Bristol Doctorate in Clinical Psychology. She previously worked at the Royal National Hospital for Rheumatic Diseases Paediatric CFS/ME service, and now works at AiR Rehabilitation Centre in Norway.

Esther Crawley is a Reader in Child Health at the University of Bristol and a Consultant Paediatrician at the Royal National Hospital for Rheumatic Diseases where she is the clinical lead for a large specialist paediatric CFS/ME service.

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