Objective Although non-invasive ventilation (NIV) can promote quality of life in motor neuron disease (MND), previous studies have disregarded the impact of progression of illness. This study explored how patients’ perceptions of NIV treatment evolve over time and how this was reflected in their adherence to NIV.
Methods Five patients with MND (male=4, mean age=59 years), from a bigger cohort who were prospectively followed, had multiple post-NIV semistructured interviews, covering more than 12 months, along with ventilator interaction data. The transcribed phenomenological data were analysed using qualitative methodology.
Results Three themes emerged: experience of NIV, influence on attitudes and perceived impact of NIV on prognosis. The ventilator interaction data identified regular use of NIV by four participants who each gave positive account of their experience of NIV treatment, and irregular use by one participant who at interview revealed a negative attitude to NIV treatment and in whom MND induced feelings of hopelessness.
Conclusions This exploratory study suggests that a positive coping style, adaptation and hope are key factors for psychological well-being and better adherence to NIV. More studies are needed to determine these relationships.
- Neurological Conditions
- Psychological Care
- Quality of Life
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Non-invasive ventilation (NIV) has been acknowledged to improve quality of life among individuals with motor neuron disease (MND).1–3 While there is a lack of consensus on how to define ‘quality of life’, it is agreed that it is multidimensional and subjective.4 To date cited improvements in quality of life through NIV treatment in MND have largely been measured quantitatively and while demonstrative of improvement, such methodology does not provide any detailed idiographic account of the patient perspective. Although a few qualitative studies have been conducted that explore experiences of the ventilator of patients with MND5–7 none have observed patient experiences longitudinally. Since NIV prolongs survival without slowing disease progression, long term users of NIV face coping with increasingly severe disability, which may alter their perceptions of NIV.
Previous studies exploring the long term experience of NIV in other diseases report positive and negative impacts. On one hand, NIV is seen to sustain life, improve physical symptoms and give active life.8–10 On the other hand, the treatment is perceived to cause inconvenience, make patients vulnerable to technology, and to serve as a reminder of physical deterioration and increased reliance on others.8–10
The primary aim of this study was to qualitatively explore how people with MND experience NIV and how this changes over time as the illness progresses. Furthermore, adherence to NIV treatment as determined by ventilator data was monitored to determine whether the qualitative findings were reflected in the pattern of equipment usage. NIV treatment benefits survival if it is used for more than 4 h per night11; this level of engagement with the treatment was used as a marker in this study.
This study was part of a larger 3-year longitudinal, prospective study of NIV treatment in MND from National Health Service (NHS) neurology and respiratory outpatient clinics in Liverpool, UK. An invitation to take part in the study was made to 35 patients diagnosed with MND based on El Escorial criteria12 at the time of referral for ventilation. Exclusion criteria included symptoms of cognitive or behavioural dysfunction. Participants were followed up every 3 months from the time of referral for pulmonary assessment until their death, or withdrawal from research.
Inclusion criteria for this study were survival on NIV treatment for more than 12 months, and the availability of continuous ventilator interaction data. Five patients from the main study met these criteria. The characteristics of the participants, including their relationship to their caregiver, are provided in table 1. All participants were living with their primary caregiver.
Semistructured interviews were employed for this study to allow the researchers to be flexible in exploring any interesting and relevant issues mentioned by participants.13 The interview schedule was developed by a multidisciplinary team: a neurology consultant, two respiratory consultants and one psychologist. The interview schedule included personal views of NIV treatment, perceived impact of NIV over time, and whether their experiences and perceptions of NIV were affected by further physical deterioration. An example of questions asked during a follow-up visit was: “How have physical changes influenced your experience of NIV?” Participants were also encouraged to talk about anything they felt relevant to the topic. Critically, the interviews aimed to explore in detail how each of the participants perceived and made sense of NIV treatment.
The interviews were all conducted by one of the researchers (HA), who was independent of the respiratory and neurology care teams. Interviews took place either at the patient's home or at hospital, depending on patient's preference. All the interviews were audio recorded and transcribed verbatim by HA and two assistants employed by the Walton Centre NHS Foundation Trust. Interviews were normally conducted verbally. However, some strategies were employed to mitigate communication difficulties with patients who had dysarthria. The need for communication alternatives was justified by Philpin et al14 when interviewing individuals with speech difficulty. Although it was preferred that the participant was alone during the interview, the carer contributed to interviews with Rosie at her request.
The number and timing of visits/interviews for each participant are listed in table 2. (All names are anonymised.) Although participants were recruited at the time of consideration for NIV use, initiation of NIV depended on results of their respiratory assessments and this resulted in variance in timing and number of interview visits. The reason for the big time gap (6 months) between the last two visits with Damien was due to his availability.
Patient-ventilator interaction data
Data on adherence to NIV was obtained at each visit from the memory card located in the ventilator (Respironics BiPAP Synchrony 2). The adherence of NIV was assessed by average days with NIV and the average usage time of the device. Participants were recommended to use NIV initially at night during sleep for at least 4 h, based on the recommended minimum time for benefit.11
Interpretative phenomenological analysis (IPA) was employed to fully describe the lived experiences of the participants. Since our primary aim was to understand the personal participation with and use of NIV over time in MND while the condition deteriorates, IPA was perceived to be the most appropriate method. IPA is interested in human experience which is studied rigorously through individual cases and this type of analysis typically involves small samples.15 ,16 Furthermore, detailed analysis of individual cases allows researchers to draw upon an individual's experiences to report shared perceptions and understandings of a common situation: long-term NIV treatment in MND.
Although the in-depth analyses were conducted by one researcher (HA), the process of how the researcher has tried to understand the participants is made transparent by the use of verbatim quotes. This accessibility of the data has been made so that the credibility of the interpretation may be examined. We accept that the interpretation presented here may not be the only way of interpreting the participant's accounts.17
The findings include clear themes which contain the participant's and the interpreter's understandings of the phenomena. That is, the analysis steps described by Smith et al15 were modelled in this study. The analysis involved familiarisation with the data, annotation of initial notes through a three-level analysis and development of themes. The themes were arranged according to their relevance to the aims of the research and to their significance in relation to reflecting participant's lived experiences. Once superordinate themes were identified, a table was created with their subthemes. Broader generalisations of the experience of NIV, in the context of MND, were afforded through exploring the patterns across the cases. Interviews were analysed without knowledge of patient-ventilator interaction data.
The study was approved by the local Research Ethics Committee for the north-west region, UK. All participants were provided with a participant information sheet that explained their involvement in the study and their future treatment. Consent was not obtained until participants had enough time to consider their participation.
Three superordinate themes emerged: experiences of NIV, influence on attitudes and perceived impact of NIV on prognosis. Each of these themes are described with an indication of their meaning for the patient and illustrated by verbatim quotes from their narratives, with longer quotes in brief sequential tables 3⇓–5. Each statement was cross referenced from the table into narrative of interpretation.
Experiences of NIV: physical-psychological relationship
Benefits of NIV were discussed from physical and psychological perspectives. With regard to the physical benefits, all participants reported improvements in their night sleep, tiredness, energy levels, shortness of breath, daily activity and oxygen levels, with the last symptom being objectively monitored and informed by respiratory clinicians. Quotes (1)a and (1)b illustrate this.
The powerful impact and the positive outcomes of NIV on physical aspects were well captured as an ‘energiser battery’ (Rosie, point 2). The benefits seen in their physical improvements were linked to positive outcomes on their psychological condition for all participants, except Stephen. For example, Oliver described how uncontrollable his sleepiness used to be before NIV. In contrast to this ‘uncomfortable’ experience of not being in control of his behaviour, Oliver described his life to have changed and become ‘more pleasant’ with NIV. Further positive psychological outcome resulting from energy improvement during the day was expressed by Rosie as ‘I can go out and enjoy it (life) not just want to sit on couch and doze” (point 2). Therefore, NIV reduces uncomfortable experience, consequently making life more pleasant, and gives individuals the opportunity to enjoy life by improving energy levels. These physical and psychological benefits were persistently perceived over time as depicted by Oliver: (1)c, (1)d, (1)e and (1)f.
Negative experiences with NIV were also observed physically and psychologically. In terms of physical aspects, issues of discomfort from the mask, strap and air pressure were mentioned by everyone apart from Mark: (1)g. Unlike benefits of NIV, however, these negative features did not seem to trigger a negative psychological response, but were accepted as a cost of NIV treatment: ‘I wouldn't want to be without it but it would be nice to have a night off” (Rosie, Point 5). The most commonly cited negative psychological experience of NIV, shared by Damien, Rosie and Stephen, was a dependence issue. Rosie, for instance argued: ‘I don't want to be vegetable’ at point 1, yet her view became one of appreciation of benefits of NIV over time. The same drift was observed with Damien. Stephen, however, consistently expressed his reluctance to the regular use of NIV, worrying that he would become dependent on NIV. His attitude at Point 4 was the same as at the start of treatment: (1)h, i.
Influence on attitudes
A link between general coping styles and the pattern of NIV use was perceived. Resistance was observed among the participants in their efforts to maintain their way of life as much as possible: “We are just carrying on doing what we've always done”(Oliver, Point 2). While resistant attitudes towards MND were persistent over time, attitudes towards MND did not determine perception or use of NIV. Instead, the following accounts made by Oliver even indicate proactive engagement with NIV to maintain his social activities, reflecting his determination to maintain his life: (2)a.
However, this pattern was not perceived with Stephen. Stephen also displayed resistance to MND: “I go to work and some days it is killing me to go. But I have to keep going and not give in” (Stephen, Point 3). The same attitude was then observed in his approach to NIV use: (2)b. In his case, the reluctant attitude towards the ventilation appeared to reflect his resistance to the illness itself; it was as if he understood regular use of NIV as surrender to the illness.
The four patients who reported psychological benefits of NIV displayed active coping styles: active engagement with problems, adaptation and acceptance. These positive coping styles were then found to reflect their attitude towards NIV. Damien displayed active problem solving attitude in general: “I mean, I've tended to look things up in advance really, you know, so I know what to expect.” (Damien, Point 1). In the same way, preparation for a possible encounter with problems was observed in his approach to the device: (2)c.
Oliver accepted his disabilities and the need of help from others, further indicating adaptation to changes: (2)d. An acceptance of NIV was also in response to perceived need for NIV overnight: (2)e.
Another prominent factor observed among the participants with positive experience of NIV was that they appreciated the life they had and affirmed their desire to stay alive: “I would like to live and living (sic) as long as I can” (Mark, point 5). Also, Damien argued for the goodness of his life: “life is good, living is, I think, better than dying” (point 4). For these patients, using NIV was important since the device was seen as a lifeline “It keeps me alive” (Mark, Point 5). Nonetheless, quality of life was also reported to be important as asserted by Rosie: “(NIV is) good for better quality. Good for bit of quantity” (point 1).
Perceived impact of NIV on prognosis
Two distinct approaches to NIV use as a treatment to alter prognosis came out of the interviews: reinforced or reluctant. The perceived essentiality of NIV underpinned the reinforced approach. Oliver described the need for NIV as “I can't envisage sleeping without it ….. I need it” (point 3). The essentiality of NIV was further reported by others in relation to a fear of death. For instance, Rosie reported that she became ‘panicky’ (point 5) if she did not have NIV at night and this was due to worries that she might stop breathing while she was asleep. Similarly, Damien explained that the potential of death resulting from his impaired respiratory function was frightening for him. He depicted the desperate situation he was in before accepting NIV: “I couldn't even use air for speaking and it was really a question of survival” (point 1).
‘Reluctance’ to use NIV in response to anticipated disability prognosis was initially observed in Damien and Rosie who expressed their unwillingness to cling onto life, yet this was reduced over time as seen in the following accounts from Damien over time: “Originally, my thoughts were to do whatever you have to do to end it when you want to, you know. And that's still probably in my thoughts” (Damien, Point 1) and (3)a. Furthermore, this change in his attitude towards life was perceived to be partly due to using NIV; it had been reported that NIV had a positive impact on social activities, enabling him to engage more with the life he came to enjoy: (3)b.
This change, however, was not observed with Stephen who attributed his depressed mood to the hopeless condition of MND: “fed up and depressed because nothing can be done” (point 2). Rather than valuing the benefits of NIV, Stephen explained: “I try to blank it. There is no cure. Why worry (about using NIV)” (point 3). His focus on the eventual fatal outcome of MND was to the detriment of evaluating the potential for amelioration of his current state.
An adaptive change in attitude towards NIV was observed where perceived benefits of the treatment exceeded their concerns over prognosis. In contrast, little shift in Stephen's reluctant engagement with NIV was related to a consistent passive approach to MND.
Patient-ventilator interaction data and an integration with the narratives
All patients, except Stephen, used NIV most nights (93.7%) over the 12 months and they used NIV for longer than the recommended time of 4 h a night:11 the mean average usage time was 9 h 27 min. Stephen, however, used NIV less that half of the nights (40.3%) during the same period and his mean usage time was only half of the recommended period of 4 h being 2 h 8 min. As the illness progressed, increased NIV usage each night was seen with Oliver, Damien and Rosie. Average usage time and percentage days with NIV for each participant over time are listed in table 6. The drop in per cent days of NIV use with Rosie at Point 4 was due to machinery failure which required a replacement while the device was serviced. No indication was made by Mark during interviews that reflect the reduced usage days of NIV seen at Point 3. The gradually increased duration of use of NIV in Rosie, Mark and Damien documented objectively was not reported by these patients in their interviews.
From these findings together with the qualitative data, it may be suggested that a focus on gains from NIV, a positive coping style and perceived need to engage with the treatment is related to better adherence. Conversely, concerns for the unknown, a negative coping style and hopelessness, is associated with poor adherence. An integration of the narratives and their adherence to NIV is provided in table 7. Names in Italics indicate a poor adherence to NIV (<4 h) and other names indicate a good adherence (>4 h).
This study explored the experience of NIV in five patients with MND who had used this treatment for over 12 months. The findings from the study indicate the influence of an individual's coping styles and perceived impact of the illness on their experience of and attitudes to NIV, and its prognosis. Individual differences in coping style were observed to be stable over time despite further worsening of symptoms, in line with previous findings that premorbid individual differences affect perceived quality of life more than disease progression.18
In the present study, physical benefits gained from NIV were direct precipitators of psychological gains, but a similar physical-psychological relationship was not observed in terms of patient's negative experiences of NIV. Nonetheless, the significance of psychological aspects in relation to NIV engagement was prominent, as previously suggested in an obstructive sleep apnoea study.19 In the current study, a more positive attitude to NIV treatment was seen in individuals who appreciated gains in energy and empowerment, while a more negative attitude was associated with perceptions of hopelessness over the future.
While a measure of resistance to MND was to some extent displayed in all the participants, individuals adhering to NIV treatment showed active coping styles such as adaptation and acceptance. The importance of flexibility in prolonged stressful circumstances has been previously recognised20 ,21; it is positively associated with better coping and negatively related to symptoms of depression and anxiety.22 ,23 Similarly, anxiety has been reported to be related to behavioural disengagement from treatments.24 In the current study, individuals who showed adaptive/flexible coping styles expressed their appreciation of, and desire for, life. In contrast, Stephen's fatalistic approach was associated with behavioural disengagement in relation to NIV usage. A good quality of life was associated with the desire to live longer, also reported by others.6 ,25 This appreciation of life meant NIV was perceived to be beneficial for most patients who thus actively engaged in their NIV treatment.
MND as a progressive illness without existing cure was observed to induce either a reinforced or reluctant approach in those who accept NIV treatment. A reinforced approach to NIV was observed when the essentiality of NIV was acknowledged and this was sometimes accompanied by a fear of death. In line with the current study, the sense of need is argued to be important for treatment engagement.6 ,26 The fear of death has been reported to be one of the determinants of initiating assisted ventilation6 and a ‘facilitator’ for adherence to NIV.19 In the current study, ‘reluctant approach’, was understood to be derived from perceived hopelessness with the illness. Hopelessness is associated with aversive behaviours25 and it has been linked with a lack of perceived purpose in life27 and little amenability of the circumstances,27 ,28 and these aspects were evidently held by Stephen. Furthermore, these suggest the influence of perceived locus of control27 ,29 and illness representations;30 perceived minimal influence over outcomes in response to overwhelming MND was observed in Stephen. The perceived hopelessness and illness representation was observed to sometimes be adaptable rather than rigid, as displayed by Damien and Rosie.
Limitations of the study and recommendation for further study
Although we believe each participant was able to fully share their lived experience of NIV, the sample was small and communication difficulties, in particular with Rosie and Mark need to be addressed and reflected in the consideration of its impact on the phenomenological approach. Inconsistent timing of the interviews and the number of visits among participants should also be noted. In particular, the last interview with Stephen was 12 months after NIV initiation, while it was 15 months for Rosie. As observed with Rosie and Damien, Stephen may have altered his attitude towards the device if he had been followed for longer.
The longitudinal experience of NIV in the context of MND was explored. An individual's experience of NIV is determined by their interpretation of the illness and its perceived impact on their future which, in turn, is influenced by coping style and attitude towards life. The current study found a positive coping style, adaptation and hope are key factors for psychological well-being and better adherence to NIV.
Although little change in coping styles was perceived in the current study, participant's level of hopelessness as a result of MND was observed to be modifiable. The improvement in participant's feelings of hopelessness then positively influenced their attitude towards NIV. Based on these findings, it is suggested that clinicians address patient's illness representation where poor adherence to NIV is acknowledged, and psychological interventions take place where such distress is acknowledged. The main focus of care, however, must remain the well-being of the patient rather than better adherence to the treatment per se.
The authors would like to give special thanks to their participants who graciously gave their time to take part in this study.
Contributors The data reported in the article was collected by HA, then analysed by HA. HA drafted the article and it was revised by BC, RMA, RC, EWT and CAY. Additional editing and amendments were subsequently made by CAY, RC and EWT. Final approval was given by CAY.
Funding This study was funded by the Motor Neurone Disease Association (UK) and the Neurological Disability Fund, Walton Centre for Neurology & Neurosurgery (grant number Young/Mar07/6026).
Competing interests None.
Provenance and peer review Not commissioned; externally peer reviewed.