Acta Psychopathologica Open Access

Keywords

Eating disorders; Anorexia nervosa; Carer skills training interventions

Introduction

Anorexia nervosa (AN) is a severe psychiatric illness typically developing at age 16 (Micali et al., 2013) [1]. Guidelines from the National Institute for Clinical Excellence recommend that most people with AN be managed on an outpatient basis (National Institute for Clinical Excellence, 2004) [2]. This means that family members are often involved in the management of the illness and frequently find themselves facing a wide range of emotional and behavioral turmoil on a daily basis. Two systematic reviews, however, have found that carers themselves find their role burdensome and distressing (Anastasiadou et al., 2014; Zabala et al., 2009) [3,4] and frequently report that they lack the information, guidance and support as to how best to address the symptoms (Haigh & Treasure, 2003) [5]. A recent qualitative study also provides rich data exemplifying the stress mothers experience when their adolescent daughters have AN (Bezance & Holliday, 2014) [6]. Caregivers express a need for support both from professionals and other parents (Bezance & Holliday, 2014; Roots et al., 2009) [6,7]. We have developed skills training materials (Experienced Caregivers Helping Others (ECHO)) for sharing skills and knowledge for caregivers (lay and professional).

The "Experienced Caregivers Helping Others (ECHO)" intervention is based on the cognitive interpersonal maintenance model of AN, and aims to help caregivers focus their own strengths and resources by using effective change strategies. The resources teach communication, fear extinction and behaviour change skills (Cairns et al., 2007; Goddard et al., 2011; Grover et al., 2011; Hildebrandt et al., 2014; Pepin & King, 2013; Rhind et al., 2014; Sepulveda et al., 2009; Treasure et al., 2008) [8-15]. An additional component of this model addresses emotionally driven behaviours such as criticism, hostility, overprotection, accommodation and enabling behaviours that can be elicited because of the difficulty of coping with AN symptoms (Schmidt & Treasure, 2006; Treasure & Schmidt, 2013) [16,17]. These are modifiable behaviours and caregivers may benefit from skills training to reduce those (Rhind et al., 2014) [13]. Caregivers are encouraged to reflect on their responses to the illness and to build awareness of how they can adapt their behaviour to have a positive effect on the trajectory of the illness. Additionally, they are taught to be role models to the person they are caring for and encouraged to engage in selfcare, adaptive coping and emotional intelligence (Macdonald et al., 2014) [18].

A recent meta-analysis of interventions for caregivers found a moderate-sized reduction in caregiver burden, distress and the ECHO intervention reduced expressed emotion and accommodating and enabling behaviors (Hibbs et al., 2015b) [19]. Moreover, in a study where the ECHO intervention was added to the inpatient treatment of adults with severe enduring AN, the patients identified positive caregiver behaviour changes (Macdonald et al., 2014) [18] and also showed improved symptomatology and quality of life at six months along with reduced bed use (Hibbs et al., 2015b) [19].

This current study is a qualitative evaluation to examine feedback about the use of the ECHO intervention in families of adolescents. A published protocol provides a detailed account of the intervention and the study design (Rhind et al., 2014) [13]. The aim of the study is to examine the experiential perspective from caregivers and their adolescent loved ones of having participated in a career skills training intervention, particularly in terms of perceived behavior change in both patient and caregiver.

Methods

Participants (n=149 patient/caregiver dyads) were part of a pragmatic three-arm multi-centre parallel group pilot randomized controlled trial (Rhind et al., 2014) [13]. Consenting caregivers of patients who met the eligibility criteria were randomly allocated to receive ECHO (E) (book and DVDs) in addition to TAU, ECHO-C (EC), (book, DVDs and ten 30-40 minute telephone coaching sessions in addition to TAU, or TAU alone). The sample included caregivers and their adolescents (age 13-21 years) with AN or ED not otherwise specified AN type (EDNOS-AN) according to the DSM-IV criteria (American Psychiatric Association, 1994) [20], recruited from 38 eating disorder outpatient services across the United Kingdom (UK). The ECHO materials and coaching were delivered from the research hub. There was no further interaction between the research center and the contributing clinical sites where usual care was delivered. Follow-up assessments included computerised self-report instruments and structured interviews for both parents and patients. Main ethics approval was granted by the Northwick Park Hospitals Ethics Committee (11/H0725/4. Site specific ethics and governance approval was granted on all participating sites and this study was adopted by the Clinical Research Network (CRN).

Inclusion criteria required at least one caregiver (typically parent/s) to participate. Caregivers were required to be identified by the patient as ‘carer’, to be currently living with the patient with the intention of living together for the following year. Other close caregivers were also encouraged to take part. “Carers” are defined by criteria of the Princess Royal Trust (www.carers.org) as someone who provides unpaid help and support to a parent, child partner, relative, friend or neighbour. Patient demographics are presented in (Table 1). A full description of methodology that includes a detailed account of Participants, Intervention and Study Design appears in (Rhind et al., 2014) [13].

	ECHO–C	ECHO	TAU
	n= 21	n= 26	n= 21
Demographics
Age (years)	16.35 (2.02)	16.66 (1.98)	17.21 (2.24)
Female	19 (90.5%)	24 (92.3%)	19 (90.5%)
Ethnic Origin
White	21 (100%)	24 (92.3%)	19 (90.5%)
Asian/Black/mixed	0 (0%)	2 (7.7%)	2 (9.5%)
Education
No Qualification	7 (33.3%)	13 (50%)	8 (38.1%)
O- level / GCSE	7 (33.3%)	6 (23.1%)	8 (38.1%)
A-level	6 (28.6%)	6 (23.1%)	3 (14.3%)
University/higher degrees	0 (0%)	0 (0%)	1 (4.8%)
Other	1 (4.8%)	0 (0%)	0 (0%)
Missing	0 (0%)	1 (3.8%)	1 (4.8%)
Patient living with caregiver	20 (95.2%)	26 (100%)	19 (90.5%)
Patient face–face contact with caregiver p/week
0–7 hours	2 (9.5%)	0 (0%)	1 (4.8%)
8-14 hours	2 (9.5%)	1 (3.8%)	3 (14.3%)
15-21 hours	2 (9.5%)	6 (23.1%)	2 (9.5%)
>21 hours	14 (66.7%)	19 (73.1%)	14 (66.7%)
Clinical characteristics
Duration of illness (months)	23.95 (21.00)	24.31 (28.56)	27.62 (26.25)
Lowest lifetime BMI	16.05 (2.14)	16.25 (2.08)	14.94 (1.63)
No. previous hospital admissions
0	14 (66.7%)	19 (73.1%)	14 (66.7%)
1	5 (23.8%)	4 (15.4%)	3 (14.3%)
2	2 (9.5%)	3 (11.5%)	3 (14.3%)
3 or more	0 (0%)	0 (0%)	1 (14.3%)

Table 1: Patient characteristics by treatment group.

Qualitative feedback was ascertained at 12 months from randomization. From the 149 patient/caregiver units, feedback questionnaires were completed by 69 patients (n=26 E; n=22 EC; n=21 TAU) and 144 caregivers (n=50 E; n=47 EC; n=47 TAU). The process evaluation questionnaires have been used in previous studies of eating disorders (Gowers et al., 2002) [21] and contained questions that related to their experience of the study and either a) their caring experience (caregivers) or b) their experience of caregiver support (patients). Patient forms asked 1) for their experience in taking part in the ECHO project and 2) their perspectives of their caregiver’s role and any changes over the past year, particularly in the period following their first assessment with the outpatient eating disorders team. Meanwhile, caregivers were asked to share their experience as a caregiver of someone who has received outpatient care for AN in term of how they have reacted, coped with and managed the situation, pre and post intervention. Copies of the feedback forms are available from the first author.

Analysis

A thematic framework approach was adopted in the analysis of the written narratives on the feedback forms. Thematic analysis is a qualitative method for identifying, analyzing and reporting patterns or themes within a dataset (Braun & Clarke, 2006) [22]. Two researchers (PM and KB) worked independently on the identification of themes. An ‘utterance’ was coded and placed in a sub-theme; when the participant changed topic, this would constitute a new utterance and sub-theme. Researcher 1 (PM) worked on the entire dataset whilst Researcher 2 (KB) worked on a sub-section (10%) within. Feedback forms were read several times and initial codes generated and incorporated into meaningful clusters before being entered into the computer software programme (Nvivo Computer Software, 1999) [23]. In devising a final thematic framework, discussions took place between the two researchers on six occasions (4 x caregivers at n=20, n=81, n=115, n=144 and 3 x patients at n=10, n= 28, n=68) via skype, email and face to face. Coding procedures were discussed, coding practices assessed and agreement obtained on emerging frameworks. After each discussion, themes were either consolidated or merged into existing themes. Descriptive labels were altered to better reflect the subject matter or deleted, if deemed irrelevant to research aims Both researchers were blind to group allocation and blinding was only lifted a) once agreement had been made with regard to final thematic framework and b) upon agreement that data saturation had been achieved.

Results

Five main themes emerged from both patient and caregiver data sets. These are depicted in Table 2 (caregivers) and Table 3 (patients). In order to preserve anonymity, any personal references to either caregiver or patient in the quotations that follow are represented by the letter ‘A’.

	Caregivers                   (n)*
	ECHO		ECHO-C		TAU
	n	%	n	%	n	%
Perceived self-change
Improvements in relationship & communication	17	34	24	51	15	32
Increased awareness, coping & understanding	22	44	13	28	14	30
Reduced anxiety, anger, guilt & hostility	11	22	17	36	5	11
Mindfulness and self-nurturing behaviours	11	22	7	15	6	13
Promoting independence & responsibility	6	12	11	23	5	11
Increased compassion & tolerance	6	12	3	6	12	26
No change	3	6	5	11	4	9
Caregiver and patient efficacy
Positive relationship	26	52	17	36	22	47
Emotional support	21	42	24	51	19	40
Perceived caregiver impact	21	42	20	43	22	47
Practical support	23	46	20	43	19	40
Use of helpful strategies & techniques	12	24	20	43	17	36
Healthy functioning (patient)	19	38	14	30	9	19
Role differentiation	4	8	8	17	3	6
Little or reduced involvement	6	12	5	11	2	4
Challenges
Negative impact on self and/or family	38	76	29	62	32	68
Practical burden of continuous care	15	30	18	38	13	28
Problematic & comorbid behaviours	17	34	12	26	15	32
Slower recovery process	17	34	13	28	12	26
Doubt, uncertainty & trust issues	10	20	9	19	12	26
Relationship problems & difficulties	5	10	7	15	7	15
No impact	5	10	3	6	2	4
Resignation & hopelessness	1	2	4	9	4	9
Sibling challenges	2	4	0	0	1	2
Coping mechanisms
Social support	23	46	28	60	24	51
Work, hobbies & non eating disorder interests	14	28	13	28	12	26
Positive mindset& self-soothing strategies	8	16	5	11	6	13
Medication, alcohol, own eating problems	7	14	4	9	4	9
Education	5	10	4	9	4	9
Spirituality	3	6	0	0	1	2
Service provision and carer guidance
Positive collaboration & experience with services	20	40	23	49	19	40
Caregiver support and education	13	26	16	34	16	34
Support from current study	6	12	29	62	5	11
Problems with treatment, team and/or system	11	22	10	21	14	30
No support	12	24	4	9	14	30
Caregiver reflections	5	10	6	13	3	6

ECHO, Experienced Caregivers Helping Others
ECHO-C, Experienced Caregivers Helping Other-Coaching;
TAU, Treatment as Usual
*represents the number of participants who report experiences that belong to that particular sub-theme

Table 2: Caregivers Themes – Total n =144 (n=50E; n= 47EC; n=47TAU).

Caregiver themes

Superordinate themes for caregivers included: 1) Perceived selfchange, 2) Caregiver and patient efficacy, 3) Challenges, 4) Coping mechanisms and 5) Service provision and caregiver guidance.

Perceived self-change

Carers spoke of improvements in relationship and communication, in particular, any change(s) noticed by carers with regards to their own attitudes, behavior or approach to the AN and patient, as well as perceived improved relationships and communication with the sufferer. There appeared to be a greater number of reports in the EC group than in E alone or TAU (E: 34%; EC: 51%; TAU: 32%). Carers also reported reduced anxiety, anger and hostility in terms of a greater sense of calmness and less maladaptive emotions, e.g. guilt, anger and hostility. Again, there seemed to be a greater percentage of these reports in the EC group (E: 22%; EC: 36%; TAU: 11%).

I think we have a close and good relationship. She is able to talk to me about her anxieties. We have learnt to do this in the past year. I have been able to give her time and space. (EC)

Learning that a calmer more measured approach works better. When she is angry be non- confrontational and take a step back (E)

There were also reports of increased awareness and understanding of AN across all groups, particularly in E, (E: 44%; EC: 28%; TAU: 30%), as well as the need to promote greater independence, i.e. reports of carers having stepped back whilst recognizing the importance of the patient being more responsible for his/her own wellbeing. There were a greater incidence of promoting greater independence amongst the EC participants (E: 12%; EC: 23%; TAU: 11%).

My understanding and knowledge has been increased by attending appointments with experts. We have learnt so much how to encourage and support someone with an eating disorder. (TAU)

I have had to learn to support in a way that shows her that I trust her to make decisions/mistakes and manage the ED herself and not ‘police’ her. (EC)

Twenty four carers also reported increased emotional intelligence over the last year, in terms of inner strength and mindfulness, i.e. looking after one's own emotional and physical wellbeing (E: 22%; EC: 15%; TAU: 13%), whilst 21 carers reported increased compassion, tolerance, kindness and patience. Interestingly, in the latter, these appeared with greater frequency in TAU (E: 12%; EC: 6%; TAU: 26%).

I know I have to protect myself (near the oxygen mask) to be of any good to A. I need to be centered/positive (E)

I am a lot more patient as a result of my sister's illness. (TAU)

Twelve participants reported no change in their behaviour or approach to the illness over the course of last year, i.e. during and after intervention.

Caregiver and patient efficacy

There were some interesting observations within this superordinate theme. Use of helpful strategies and techniques, for example, included many of the skills taught in the in the intervention materials, such as communication techniques, calmness, openness and honesty, passing more responsibility to sufferer for wellbeing, bigger picture thinking and challenging comfort zones. It was interesting to note that although many of the skills and techniques referred to are taught in the New Maudsley approach, references to the skills appeared in the narratives of all three groups, including TAU which might suggest a "contamination" effect in TAU (E: 24%; EC: 43%; TAU: 36%). The following three quotes, for example, were all written by caregivers in TAU and one quote in particular appears to imply previous training in caregiver skills.

Communicating without conflict and accusation (from workshops done) and understanding that some behaviours are normal when dealing with anorexia. (TAU)

Externalizing eating disorder, not getting drawn into discussions on food. Sticking rigidly to food plan.(TAU)

Remember that the moods (outbursts) are the disorder and not daughter. Listen to her feelings and try to discuss worries. (TAU)

The sub-theme ‘healthy functioning (patient)’ contained the researchers’ interpretations of narratives that indicated normalized eating behaviours and the patient engaging in normal life with regards to school, social life and healthier eating behaviour. Although in some cases, narratives contained some signs of AN behaviours present, in general, feedback indicated a normal age related lifestyle with little support required from caregivers or professionals. There was a higher incidence of these reports in the intervention groups (E: 38%; EC: 30%; TAU: 19%). Although one should exercise caution in directly attributing any change to intervention effects, questions posed in the feedback forms did relate to changes ‘during the past year’, i.e. between the pre and post intervention periods.

He's doing really, really well. Working towards recovery. Doing well at university, come on a lot in the past year, gone for an interview for a job today, lots more confidence. (E)

Coping well. Eating normally, still on anti-depressants but free in the main from crippling anxiety – hoorah! (EC)

Over 40% (n=65) of the sample reported sharing a positive relationship with their loved one, despite the challenges of living alongside AN. Carers also reported providing emotional support, (encouragement, guidance, compassion) and practical support (mealtime support, financial support, transportation, food shopping, shared goal setting and problem solving strategies) with a slightly higher incidence of emotional support being reported in the guided self-help group (E: 42%; EC: 51%; TAU: 40%) .

Being there for her, not being judgmental. Gently persuading her to eat. Reminding her that she is loved by so many people and that her friends adore her. (EC) …

Had to keep a diary for his eating…had to eat with him at a set time, made sure he had his breakfast, lunch and tea put in front of him … (E)

Caregivers across the sample also reported an awareness of caregiver impact, with narratives reflecting on the consequences and influence of one’s own actions on the trajectory of the illness.

You have to be so careful about saying the right thing. The person affected is in such a vulnerable place in their lives that you do need to filter what you say, particularly regarding the disorder. (TAU)

Caregiver challenges

Sixty nine percent of caregivers (n=99) described the negative impact on self and/or family of living alongside AN. Narratives included reports of helplessness, frustration, high anxiety, hostility, conflict, anger, guilt, exhaustion and fear, as well as disruptions to family life in general. There were several reports of marital and relationship discord, feelings of isolation and concern for siblings, as well as detrimental consequences on one’s own mental and physical health.

Particularly hard on brother and sister. They found it difficult to be at home (both living away from home). Sister particularly found it more stressful – unsure what to say to her. She also felt uncomfortable around her – body image, weight issues. She felt A was ‘judging’ her/ comparing herself with her. (E)

Marriage breakdown….trying to maintain stable home life for myself, A and son. (EC)

There have been months where I have been powerless to stop/slow down A’s decline. It has been terrifying. Our worst year. I have felt alone and overwhelmed. Useless. (T)

Practical burden of continuous care, i.e. balancing the demands of care with other life commitments, including financial burden, was reported by almost a third of the sample. Likewise, problematic and comorbid behaviours, reflected the existence of continued AN behaviours, denial, maladaptive thoughts and emotional turmoil along with refusal to engage in therapy, exclusion of caregivers in any form of attempted support, scant participation in healthy life activities, i.e. little improvement in eating or other comorbid behaviours.

We have had to call the police and paramedics twice because she became uncontrollable and they had to restrain her. We had also had to deal with A self-harming or attempting to self-harm. (EC)

Twenty nine percent of the carers (n=42) referred to the protracted course of the illness whilst 21% of the sample (n=31) wrote of their doubts, uncertainties and trust issues, including self-doubt in their own ability as ‘caregiver’, along with fear for the future and continuous struggles to understand the illness, their caring role and its impact.

She is coping in a calmer manner with her situation with less extreme behaviours. Her weight is now stable but she still becomes distressed by this. Her aim is to be back at school full time and she is making progress with this although sometimes with difficulty. (EC)

Fears even when things are OK that we’re missing something and it won’t be OK for long. (TAU)

Weaker sub-themes included relationship problems and difficulties, carers feeling they had no impact whatsoever on their loved one’s mindset and behavior, resignation and hopelessness and sibling challenges.

I don't think I have an impact on A, she is her own person, an adult as she keeps reminding me. (E)

Coping Mechanisms

The data generated interesting findings on carer coping mechanisms; the most widely reported being social support and work, hobbies and non-eating disorder interests.

I gain support from my daughters, siblings and sometimes (more so latterly) from my partner – her father. (EC)

I have kept working which has helped me enormously to continue to have a life outside of the home. (TAU)

Other coping mechanisms included positive mindset and selfsoothing strategies (the use of emotional intelligence and positive thinking strategies), education (reading of literature, surfing the web, active searching for information on EDs and mental illness), medication, alcohol and own eating problems used to dull emotional pain and spirituality (the use of prayer or one’s faith as a coping strategy).

It’s been hard to watch her at her lowest psychological point, but I’ve always tried to stay positive. (TAU)

Part of my coping strategy has definitely involved alcohol, I am afraid to say. Some evenings involve several glasses of wine to blank out the past and to induce sleep. Not a great solution, I admit, but unfortunately the truth. (E)

Most helpful is the internet – reading about anorexia, research evidence. (EC)

Service provision and caregiver guidance

Sub-themes within the final superordinate theme include aspects of service provision, i.e. support from Eating Disorder Units, General Practitioners as well as independent support groups, research projects or individual counselling received. Forty-three percent (n=62) of the caregivers described positive collaboration and experience with the treatment team (E: 40%; EC: 49%; TAU: 40%).

The impact of outpatient care was huge. I was terribly worried about A and great relief knowing that we could get help. The regular meetings and advice helped greatly. There was an orderliness about the routine, step by step advancement to getting A to a better place. (EC)

Just under a third of participants, spread evenly across the groups, reported some form of caregiver support and education, such as attending counseling, therapy, caregiver training, caregiver support groups. In some cases, this was offered through the service provider whilst others sought guidance independently.

We felt empowered to help by the support group and the strategies they gave us worked for our daughter. Thank you to all the team. (TAU)

Twenty eight percent (n=40) of the sample made reference in some form or other to the current study. Narratives related to the DVDs and/or book, or indeed, disappointment at not having received support.

I would have loved some support! As I was on the ‘no help’ strand of the study I can only hope it is better for stress. (TAU)

A quarter of the sample (E: 22%; EC: 21%; TAU: 30%) expressed dissatisfaction with the treatment, team or the system, either from the patient or caregiver’s perspective. Examples include criticism of treatment given, or aspects of the team itself, nonadherence to care or personal frustration over lack of knowledge.

I find the argument of confidentiality difficult, one day we have a level of communication, the next it changes, despite people talking about a collaborative approach and my daughter expressing that she is happy for there to be open communication. (TAU)

The final two sub-themes were caregiver reflections over the illness or caring role, in general. There were also accounts of having received no support whatsoever (E: 24%; EC: 9%; TAU: 30%).

Getting back to normal after the very little support we received became absolutely none. (TAU) (Table 2).

Patient Themes

Superordinate themes for patients included: 1) Perceived changes in caregiver(s) 2) Helpful factors towards recovery 3) Unhelpful caregiver behaviours 4) Experiential perspective of study participation and 5) Relationship with caregivers.

Perceived change in caregiver(s)

These narratives represented patient perceptions of any changes they had noticed in their caregiver(s) over the course of the last year, i.e. pre and post intervention periods. Forty five patients reported greater insight and confidence in their carers, the ability to challenge problematic behaviours as well as improved understanding and awareness with suggestions of improved coping and coping strategies. Reports of greater understanding, awareness and coping were more frequent in the intervention groups (E: 73%; EC: 76% and TAU: 48%).

My Mum (primary carer) has a very good understanding of the illness. This has improved immensely since being involved with this study. (EC)

There were also reports of improved relationship and communication in terms of less distance, more openness and honesty in relationships and greater shared participation in noneating disorder communication and activities. Again, narratives appeared more in the intervention groups with the greatest frequency occurring in E (E: 42%; EC: 29%; TAU: 19%). Twenty participants reported a calmer behavioural and emotional climate in terms of calmer family dynamics, i.e. less anger, stress and anxiety along with increased patience in their carers. Likewise, these reports occurred more regularly in the intervention groups (E: 35%; EC: 33%; TAU: 19%).

She now talks to me about her concerns and worries involving my illness. More honest and open and is more understanding and less judgmental. All this means that she is able to know what support I need (emotionally or otherwise) (EC)

My Mum also remains a lot calmer now, as she seems to feel less responsible for my eating. Previously my Mum had a tendency to overreact at the least thing – she seems to have this more under control now. (E)

Interestingly, feedback also highlighted contamination effects, in that some caregivers in TAU group appeared to have had access to the New Maudsley materials.

She has done a lot of reading around the disorder too, including several of Janet Treasure's books and those released by the Maudsley. Over the past year, she has identified (more so than I) that perhaps many of my most rigid habits around food are less induced by AN but instead by OCD and ASD. (TAU)

Helpful factors towards recovery

Patients offered a wide range of factors they deemed helpful in the recovery process. Over half the patients, for example, listed open and non-judgmental communication and honesty with the use of listening and questioning strategies as an important contributor to the recovery process.

Talking and listening to each other in a calm, open manner is very helpful. (EC)

Monitoring of meals and food intake in terms of preparation, buying food and meal support was also considered important to recovery.

Having my dinner ready when I get home from school so I cannot binge and monitoring me after dinner to make sure I am not sick. (E)

Motivational support, i.e. awareness, understanding, praise and encouragement and distractions and normality, including bigger picture thinking, non-eating disorder talk and activities were regarded important by a quarter of the sample.

She has never become angry or irritated which has been very helpful and has always been understanding and patient which has made my life easier. (EC)

Distractions and normality, i.e. bigger picture thinking, including participation in non-eating disorder activities and conversation were thought to be helpful by almost a quarter of the sample.

I find it helpful when we talk normally and when we make meal times normal so they don’t seem like a big deal. (E)

Unhelpful caregiver behaviours

High expressed emotion was the most commonly quoted factor impeding recovery. Anger, anxiety, blame, stress, hostility and criticism is cited by 40% (n=27) of the sample. This factor appeared more frequently in the narratives in the intervention groups (E: 46%; EC: 43%; TAU: 29%). It is interesting that there was a higher frequency of E and EC patients’ observations of a calmer behavioural and emotional climate in ‘Perceived change in caregiver’. This may be further indicative of a certain element of change having occurred. Weak boundaries and accommodating to symptoms were also deemed unhelpful by a third of the sample.

However, my mum does not know how to handle it when I refuse to eat and gets easily frustrated, angry and upset. I do not find this helpful – it makes it more difficult for me. (E)

Aggression is not helpful their frustration and them trying to make me just deal with situations they throw on me without thinking how this may dislodge my current progress. (TAU)

She even buys food from multiple different shops so that she can guarantee I’ll eat. (EC)

Lack of physical or emotional presence and communication problems were also regarded as unhelpful. Caregivers at work or living away from home allowed the patient to lapse into problematic behaviours. Lack of emotional presence, e.g. neglecting behaviours due to other life commitments was also difficult for the patient. This are links here to communication problems and misunderstandings, lack of understanding or parents ignoring behaviours in a bid to keep the peace.

They did not let it thrive although if they were busy I had the opportunity to play into the hands of the illness without them realizing. (E)

Persistent questioning or assumptions that I will “go and have a carrot” are not helpful. (TAU)

The remaining factors patients report as detrimental to recovery include food, weight and shape issues and talk, lack of understanding, intrusiveness and control, disclosure of illness to others and one patient describes her caregiver’s own eating and food issues as unhelpful.

When negative comments are made about their shape or others' shape, makes me question my own more. (E)

I sometimes found that the more rules and barriers my Mum tried to implement, the more determined my eating disorder would be to fight against them. Particularly if our arguments became very verbal, the stubbornness of my eating disorder would thrive. (EC)

She is often dieting and discussing with me her body hatred. She told me I looked lovely when very ill which I misinterpreted. (TAU)

Experiential perspective of study participation

Seventy eight percent of participants (n=53) reported positive aspects of having taken part in the study (E: 92%; EC: 81%; TAU: 57%). Narratives reflect appreciation, acceptability and positive impact of study, i.e. acceptability of questionnaires and monthly telephone calls, or a general feeling of the project having helped their situation or that of their caregiver by enhancing selfreflection.

The extra information has been brilliant. Giving my carers the information they needed to cope and to support me through the illness. (E)

Meanwhile, a similar proportion of the sample, 75%, (n=51) reported negative aspects of participation in terms of problems with telephone calls, difficulties encountered in completing questionnaires and of the study having had a negative impact on either self or caregiver. Examples included the emotional impact of being reminded of AN symptoms in the maintenance period, or the feeling of having been duped into participating and not receiving any help (TAU). There was also some confusion, particularly after outpatient treatment had ended, as to what period of time the questions referred.

Felt that myself and my parents had been 'conned' by my ED nurse into participating, We thought we would be getting help and advice but ended up being the group that got nothing. (TAU)

If completely honest, I didn’t have any positive experience from this project. As I recovered very quickly the project was completely out of sync with my personal recovery and should have been kept alongside each person’s personal recovery time, rather than using an ‘average’ recovery sign. (E)

to be honest I didn’t get anything out of this. There was nothing really there to help me – it was just questionnaires (EC).

Thirty nine patients felt that the intervention had no perceivable effects on either themselves or their carers. Interestingly, there was a higher percentage of patients in E who reported no particular intervention effects (E: 73%; EC: 43%; TAU: 52%).

It hasn’t changed I don’t think – carer #1 is none the wiser as that is how she deals with things. Carer #2 did his own research and didn’t really have anything to do with this project. (E)

Meanwhile, 25 out of the 68 patients gave altruistic reasons for participation. Many cited feeling a sense of 'giving back' and the desire to help others. These reports occurred with greater frequency in the intervention groups, particularly in the EC group (E: 35%; EC: 52%; TAU: 24%).

I felt encouraged that I was able to be useful in some way and contribute towards a programme designed to improve the lives of carers and those suffering with an eating disorder. (EC)

Relationship with caregivers

The final superordinate theme contained sub-themes referring to relational aspects. Seventy three percent of patients (n=50) expressed an appreciation of caregiver impact, support and understanding in the manner they approach and deal with the illness. They reported caregiver coping strategies and appreciation of constant support. Sixty six percent of the sample (n=45) also reported aspects of a close relationship with their caregiver.

Even though I shield them from the ED and my low moods as much as possible, it is highly beneficial to know that they are there and that their love, care and support is ongoing and unconditional. Without such support, I would have no defense against the ED onslaught and would simply bow down to it. (TAU)

However, almost half the sample also (n=33) reported problematic aspects of the relationship to varying degrees, which is understandable given the stressful conditions of living alongside AN. Narratives indicated a lack of understanding, problematic caregiver coping, caregiver anxiety and belittling of the illness.

The expectation for them of recovery is for me to like who I am and whatever I look like. My mum especially believes that people in the healthy weight range do not have such intrusive thoughts – she doesn’t understand that mental illness cannot be fixed by weight gain. (TAU)

There were also some suggestions of patients experiencing feelings of guilt, empathy and awareness of caregivers’ struggles (n=22; 32%). Guilt is often particularly pronounced when the patient recognizes the impact of their illness on their caregiver’s own mental or physical wellbeing.

I think mum finds it hard always trying to keep the peace. She now has high blood pressure and diabetes…. I worry that my anorexia has been detrimental on her health. Dad struggles to cope with and understand the anorexia. (EC) (Table 3)

			Patients (n)*
	ECHO		ECHO-C		TAU
	n%		n%		n%
Perceived changes in caregiver(s)
Greater understanding, awareness & coping	19	73	16	76	10	48
Improved relationship & communication	11	42	6	29	4	19
Calmer behavioural & emotional climate	9	35	7	33	4	19
Greater independence & trust	1	4	2	10	2	10
Negative changes	1	4	0	0	1	5
Helpful factors towards recovery
Adaptive communication & honesty	11	42	11	52	13	62
Monitoring of meals & food intake	9	35	10	48	7	33
Motivational support	8	31	3	14	7	33
Distractions & normality	9	35	4	19	3	14
Challenging behaviours & boundary setting	2	8	2	10	7	33
Calmer emotional climate	3	12	3	14	2	10
Independence & responsibility	4	15	2	10	1	5
Professional help	4	15	1	5	0	0
Concern for caregivers	0	0	1	5	1	5
Role modelling	0	0	0	0	1	5
Unhelpful caregiver behaviours
High emotion	12	46	9	43	6	29
Weak boundaries & accommodating symptoms	7	27	7	33	8	38
Lack of physical or emotional presence	9	35	1	5	4	19
Communication problems	3	12	5	24	5	24
Food, weight & shape issues/talk	6	23	1	5	2	10
Lack of understanding	5	19	2	10	2	10
Intrusiveness & control	2	8	3	14	2	10
Disclosure of illness to others	2	8	2	10	1	5
Caregivers’ own eating & food issues	0	0	0	0	1	5
Experiential perspective of study participation
Positives	24	92	17	81	12	57
Negatives	20	77	17	81	14	67
No effect on self or caregiver	19	73	9	43	11	52
Altruism	9	35	11	52	5	24
Uncertainty over study aims & methodology	1	4	4	19	1	5
Relationship with caregivers
Appreciation of caregiver impact & support	18	69	14	67	18	86
Close relationship	13	50	15	71	15	71
Problematic aspects of relationship	15	58	10	48	8	38
Guilt, empathy & awareness of carer struggles	9	35	7	33	6	29
Constructive caregiver coping approach	3	12	3	14	3	14

ECHO, Experienced Caregivers Helping Others
ECHO-C, Experienced Caregivers Helping Other-Coaching
TAU, Treatment as Usual
*represents the number of participants who report experiences that belong to that particular sub-theme

Table 3: Patient themes - Total n = 68 (n=26E; n=21EC; n=21TAU).

Discussion

This current study is a qualitative evaluation to examine feedback about the use of a caregiver skills training intervention in families with adolescents with AN. The Medical Research Council’s criteria in terms of development and evaluation of an intervention considers a) the practical effectiveness of the intervention (does it work) and b) what are the active ingredients and how are they exerting their effect (how it works). This study, then, considers the second criterion by offering a greater insight into the nature of both patients’ and carers’ needs and experiential perspectives of having taken part in a skills training intervention. Quantitative analyses have been conducted in tandem with the qualitative element (Peynenburg et al., 2015; Rhind et al., 2015) [24,25]. It is hoped that both approaches will provide a sense of complementarity, where different methods are used to assess different components. The current study examined the experiential perspectives in narratives from both patients and their caregivers after having taken part in a carer skills training intervention, particularly in light of any perceived changes reported during the post intervention period.

Caregivers

The additional coaching element was associated with a greater level of perceived self-changes, e.g. relationship improvements, reduced anxiety, anger and hostility, promoting independence and responsibility than self-help alone and this, in turn, was greater than in Treatment As Usual. Overall, there was very little feedback that suggested that the intervention was irrelevant or inappropriate for this patient group. Indeed, some caregivers said that the information would have been helpful at an even earlier stage. This is interesting as we thought that we were ascertaining patients early in their illness. Previous work has found that mothers find being given skills and practical advice helpful early in the illness (Bezance & Holliday, 2014) [4]. It is also important to consider those caregivers’ recommendations that guidance and support be given upon diagnosis and introduced at the outset of the illness. There can be a long interval between when the first symptoms develop; acknowledgment that help and accessing expert help is needed (12 months) (B-EAT, 2015) [26]. A German study also found that the duration of untreated illness in the early onset group was high (Neubauer et al., 2014) [27]. One should bear in mind the costs of such delays and the ramifications on caregivers’ (and siblings’) family and social lives. There are implications in areas of work, extra financial burden and the family members’ own physical and mental health. Delays can also carry heavy costs for their loved one’s welfare in terms of emotional and physical wellbeing, education and social development and a poorer response to treatment (Treasure et al., 2014) [28]. Finally, it was interesting to note that reports of increased compassion and tolerance were greater in Treatment As Usual than in either of the intervention groups. It may be the case that carers become accepting and acquiescent whilst living alongside AN, whereas carer interventions focus on addressing symptoms within the spirit of emotional intelligence, by using communication techniques, challenging comfort zones, promoting responsibility and using consistent non-negotiable boundaries. Further research would be required to examine this point further.

Patients

Overall, there appears to be a higher frequency of patients reporting greater understanding, awareness and coping, improved relationships and a calmer emotional and behavioural climate in the intervention groups (E and EC) over Treatment As Usual. This is consistent with ethos taught by the ECHO skills training interventions (Goddard et al., 2011; Pepin & King, 2013; Goddard et al., 2010; Hibbs et al., 2015a;) [9,12,29,30]. Results are also consistent with a previous phase of our work where we compared the ECHO intervention with Treatment As Usual in adult patients and their caregivers. It is useful to compare feedback from the two groups in terms of providing relevant information and skills for future intervention development. Again, patients’ perceptions of greater understanding, awareness and coping abilities, improved relationships and communication and reduced criticism, overprotection and carer anxiety was more frequent in the intervention group (Macdonald et al., 2014) [15].

It is also interesting to note that when comparing the feedback of adolescents in the earlier stage of AN with our previous work using the intervention with people at a severe, enduring phase of illness, higher endorsement of the intervention was found from both patients and caregivers in the adult group (Macdonald et al., 2014) [15]. Negative aspects of participation, for example, were reported with greater frequency in the intervention groups. It is also possible that the motivational style of coaching may not have matched the needs of caregivers in the early phase of illness. Bill Miller, for example, suggests that motivational interviewing is not appropriate when people are in action (Miller & Rollnick, 2009) [31]. For example, one caregiver did not find the reflective element helpful. It may be the case that a more directive CBT type of coaching might be more beneficial in this group. Meanwhile, some caregivers reported additional needs that were not covered, such as the possible need to consider the overlap with autistic spectrum traits. An individually tailored caregiver intervention would involve working initially on a needs assessment, discussion of change objectives before developing a practical plan, along with adoption and implentation.

Limitations

The main limitation was the use of a written feedback approach as opposed to a semi-structured interview which would have made it possible to clarify those responses of a more ambiguous nature, as well as eliciting richer and more meaningful data. Another limitation was that only half of the patients in the study provided feedback compared to the higher number of carers who provided data which may have led to a bias towards those who were particularly engaged in the intervention. There may also have been a time lapse between outpatient attendance and receipt of feedback response form. A number of patients, for example, experienced confusion over what particular time period to which the questions referred. Finally, this feedback highlighted problems in the design of the study. It appears that the information given to patients about the study was considered to be unhelpful or incorrect and some participants felt coerced. Also, the background level of dissemination of the basic aspects of the intervention is quite high with caregivers in all groups attending workshops and accessing the materials. Consequently, this appears to have resulted in some contamination in the Treatment As Usual group.

Strengths

The main strength of this study is that, as far as we are aware, it is the first study to have explored in detail, the experience of adolescent outpatients and their caregivers’ participation in a skills training intervention. Another strength was the reasonably high return of caregiver feedback questionnaires. Furthermore, it is possible to compare experiential perspectives across stage of illness between younger and older patients as the same intervention was used and measured in earlier phases of the research (Macdonald et al., 2014) [15]. Finally, blinding of assessors is also strength in terms of study methodology.

Conclusion

Changes in caregiver behaviors and attitudes are noted both by caregivers and patients, when caregivers have access to the skills training intervention. We did not find major differences in the relevance of these materials for people in the early versus severe and enduring stage of illness. The background level of caregiver support and information is greater in the adolescent group than in our study of adults but this may be because of a general increase in dissemination rather than being related to the age of the participants. However, some comments suggest that the timing and pace of the intervention may need to be different in the adolescent group.

This study ilustrates how caregiver skills training materials are successfully used by caregivers to change their behaviours. These changes in style are observed by patients as well as by caregivers themselves. Moreover, this approach can be easily disseminated and may play an important role in breaking some of the barriers to early intervention in this patient group.

Competing Interests

JT is an author of the book used in the ECHO intervention (Treasure et al., 2007). RH, CR, EG, PM and GT provided coaching in the ECHOc treatment arm.

Acknowledgements

We thank the experienced coaches who are taking part in this study: Snita Ahir, Leigh Best, Yvonne Boughton, Russell Cooper, Suzi Doyle, Jocelyn de Guzman, Amy Harrison, Jenny Langley, Esther Ritchie, Yvonne Round, Jilly Shipway, Melinda Waldrum and Clare Walker.

We also thank the principal investigators who are involved in the recruitment of participants at outpatient sites (S. Clark-Stone, 2gether NHS Foundation Trust; W. Clarke, Anuerin Bevan Local Health Board; D. Robertson, Birmingham and Solihull Mental Health NHS Foundation Trust; N. Dawson, Bradford District Care Trust; C. Schrieber-Kounine, Avon and Wiltshire Mental Health Partnership NHS Trust; J. Shapleske, Cambridgeshire and Peterborough NHS Foundation Trust; J. Whittaker, Central Manchester University Hospitals NHS Foundation Trust; A. Jones, Cornwall Partnership NHS Trust; K. Goss, Coventry & Warwickshire Partnership NHS Trust; H. Crowson, Derbyshire Mental Health Services NHS Trust; K. Higgins, Dudley and Walsall Mental Health NHS Trust; N. Green, Humber Mental Health Teaching NHS Trust; A. Lodwick, Hywel dda Health Board; N. Jacobs, Kent and Medway NHS and Social Care Partnership Trust; C. Newell, Kimmeridge Court, Dorset Healthcare University NHS Foundation Trust; J. Morgan, Leeds Partnership NHS Foundation Trust and St George’s University of London; J. Arcelus, Leicestershire Partnership NHS Trust; H. Birchall, Lincolnshire Partnership NHS Foundation Trust; R. Thompson, Norfolk and Waveney Mental Health Foundation Trust; H. Stephens, North Bristol NHS Trust; I. Lea, North Essex Partnership Foundation NHS Trust; L. Addicott, Nottinghamshire Healthcare NHS Trust; S. Sankar, Northamptonshire Healthcare NHS Trust; J. Holliday, Oxford Health NHS Foundation Trust; B. Waites, Powys Local Health Board; H. Strachan, Royal Bolton Hospital; A. Fennell, Black Country Partnership NHS Foundation Trust; A. Wolton, Somerset Partnership NHS Foundation Trust; H. Gahan, South Essex Partnership University NHS Foundation Trust; G. Moss, Sheffield Children’s Hospital; J. Orme, Sheffield Health and Social Care NHS Foundation Trust; K. Moore, South Staffordshire and Shropshire Healthcare NHS Foundation Trust; G. Burgoyne, Suffolk Mental Health Partnership NHS Trust; I. Eisler, South London and Maudsley NHS Foundation Trust; B. Bamford, South-West London and St George’s Mental Health Trust; I. Yi, Surrey and Borders Partnership NHS Foundation Trust; P. Parker, Worcestershire Mental Health Partnership NHS Trust).

Our appreciation extends also to the Mental Health Research Network who have been instrumental in the successful setup of this project. Finally, we gratefully acknowledge the Psychiatry Research Trust for supporting the PhD Scholarship of one of the lead researchers for this Pilot Trial (C.R.).

This manuscript presents independent research funded by the National Institute for Health Research (NIHR) under its Research for Patient Benefit programme (PB-PG-0609-19025). JT and US receive salary support from the National Institute for Health Research (NIHR) [Mental Health Biomedical Research Centre and/or Dementia Biomedical Research Unit] at South London and Maudsley NHS Foundation Trust and King's College London. The views expressed in this article are those of the authors and not necessarily those of the NHS, the NIHR or the Department of Health.

References

1. Summerskill WHJ, Davidson EA, Sherlock S, Steiner RE(1956) The neuropsychiatric syndrome associated with hepatic cirrhosis and an extensive portal collateral circulation, Q. J. Med 25:245-266.
2. Cordoba J (2014) Hepatic Encephalopathy: From the Pathogenesis to the New Treatments, International Scholarly Research Notices (ISRN) Hepatology 236268:1-16.
3. Fischer R and Maier O (2015) Interrelation of Oxidative Stress and Inflammation in Neurodegenerative Disease: Role of TNF, Oxidative Medicine and Cellular Longevity 610813:1-18.
4. Kang D and Hamasaki (2002) N Maintenance of mitochondrial DNA integrity: repair and degradation, Curr. Genet 41:311-322.
5. St-Pierre J,Buckingham AJ, S.J. Roebuck, Brand MD(2002) Topology of superoxide production from different sites in the mitochondrial electron transport chain, J. Biol. Chem 277:44784-44790.
6. Najjar S,Pearlman DM, Alper K, Najjar A and Devinsky O (2013) Neuroinflammation and psychiatric illness, J. Neuroinflammation 10:43.
7. Sriram K, Shankar SK, Boyd MR, Ravindranath V (1998) Thiol oxidation and loss of mitochondrial complex I precede excitatory amino acid mediated neurodegeneration, J. Neurosci 18:10287–10296.
8. Nersesova LS (2011) Role of creatine kinase and its substrates in the central nervous system in norm and in various pathologies, Journal of Evolutionary Biochemistry and Physiology 47:120-127.
9. Zhang SF, Hennessey T, Yang L, Starkova NN, Beal MF, et al. (2011)  Impaired Brain Creatine Kinase Activity in Huntington's Disease,Neurodegenerative Dis 8:194–201.
10. Lowry OH, Rosebrough NJ, Farr AL, Randall RJ (1951) "Protein measurement with the Folin phenol reagent", J. Biol. Chem 193:265–275.
11. Reitman S, Frankel S, Amer J, et al. (1957) A coloremetric method for the determination of serum glutamic oxaloacetic and glutamic pyruvic transamination. Am. J. Clin. Path.28 56: 56-63.
12. Doumas B, Watson W, Biggs H(1971) Albumin standards and measurment of serum albumin with bromocresol green. Clin. Chem. Acta 31:87-96.
13. Henry R(1964) Clinical chemistry, Principles and Technics, Harper& Row publisher, New Yourkp 108.
14. Misra HP and Fridovich I(1972) The role of superoxide anion in the autoxidation of epinephrine and a simple assay for superoxide dismutase, J. Biol. Chem 247:3170-3175.
15. Baudhuin P, Beaufay H, Rahman-Li Y, Sellinger OZ, Wattiaux R, et al. (1964) Tissue fractionation studies 17. Intracellular distribution of monoamine oxidase, aspartate aminotransferase, alanine aminotransferase, D-amino acid oxidase and catalase in rat-liver tissue, Biochem.J 92:179–184.
16. Pinto REand Bartley W (1969) The nature of the sex-linked differences in glutathione peroxidase activity and aerobic oxidation of glutathione in male and female rat liver, Biochem. J 112:108-115.
17. Moron MS, Depierre JW, Mannervik B (1979) Levels of glutathione, glutathione reductase and glutathione S-transferase activities in rat lung and liver, Biochem. Biophys. Acta 582:67-78.
18. Cini M, Fariello RG, Bianchetti A, Moretti A (1994) Studies on lipid peroxidation in the rat brain, Neurochemical Research 19:283-288.
19. Hughes BP(1962) A method for estimation of serum creatine kinase and its use in comparing creatine kinase and aldolase activity in normal and pathological sera, Clin. Chim. Acta 7:597–603.
20. Palmer JW, Tandler B, Hoppel CL (1977) Biochemical properties of subsarcolemmal and interfibrillar mitochondria isolated from rat cardiac muscle, J. Biol. Chem 252:8731-8739.
21. Bogenhagen D and Clayton DA(1974) The number of mitochondrial DNA genomes in mouse L- and human HeLa cells: Quantitative isolation of mitochondrial DNA, J. Biol. Chem 249:7991-7995.
22. Lambert AJ and Brand MD (2004) Inhibitors of the quinone-binding site allow rapid superoxide production from mitochondrial NADH:ubiquinone oxidoreductase (complex I), J. Biol. Chem 279:39414-39420.
23. Smeitink J, Sengers R, Trijbels F, van den Heuvel L (2001) Human NADH:ubiquinone oxidoreductase, J. Bioenerg. Biomembr 33:259-266.
24. Murphy CR, Rama RKV, Bai G, Norenberg MD (2001) Ammonia-induced production of free radicals in primary cultures of rat astrocytes, J. Neurosci. Res 66:282-288.
25. Cichoz-Lach H and Michalak A (2014) Oxidative stress as a crucial factor in liver diseases, World J Gastroenterol20:8082–8091
26. Chen X, Guo C and Kong J (2012) Oxidative stress in neurodegenerative diseases, Neural Regen Res7:376–385.
27. Ghezzi D and Zeviani M (2012) Assembly factors of human mitochondrial respiratory chain complexes: physiology and pathophysiology, Adv Exp Med Biol 748:65–106.
28. Wei YH, Lu CY, Wei CY, Ma YS and Lee HC (2001) Oxidative stress in human aging and mitochondrial disease? consequences of defective mitochondrial respiration and impaired antioxidant enzyme system, Chin J Physiol44:1–11.
29. Selivanov VA, Votyakova TV, Pivtoraiko VN, Zeak J, Sukhomlin T, et al. (2011) Reactive oxygen species production by forward and reverse electron fluxes in the mitochondrial respiratory chain, PLoS Comput Biol 3:e1001115.
30. Finkel T, Holbrook NJ (2000) Oxidants, oxidative stress and the biology of ageing. Nature 6809:239–247.
31. Chadipiralla K, Reddanna P,Chinta RM, Reddy PVB (2012) Thioacetamide-Induced Fulminant Hepatic Failure Induces Cerebral Mitochondrial Dysfunction by Altering the Electron Transport Chain Complexes, Neurochemical Research 1: 59-68.
32. Boveris A, Cadenas E (2000) Mitochondrial production of hydrogen peroxide regulation by nitric oxide and the role of ubisemiquinone, IUBMB Life 50:245–250.
33. Sriram K, Shankar SK, Boyd MR, Ravindranath V(1998) Thiol oxidation and loss of mitochondrial complex I precede excitatory amino acid mediated neurodegeneration, J. Neurosci 18:10287–10296.
34. Streijger F, Oerlemans F, Ellenbroek BA, Jost CR, Wieringa B, et al. (2005) Structural and behavioural consequences of double deficiency for creatine kinases BCK and UbCKmi, Behavioural Brain Research 157:219–234.